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Bilal Kirmani, Basitt Kirmani, Franco Sogliani, Should asymptomatic bronchogenic cysts in adults be treated conservatively or with surgery?, Interactive CardioVascular and Thoracic Surgery, Volume 11, Issue 5, November 2010, Pages 649–659, https://doi.org/10.1510/icvts.2010.233114
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Abstract
A best evidence topic in cardiac surgery was written according to a structured protocol. The question addressed was whether asymptomatic bronchogenic cysts in adults require surgery or whether they can be adequately managed with conservative treatment or observation only. Altogether more than 310 papers were found using the reported search of which 23 represented the best evidence to answer the clinical question. The authors, journal, date and country of publication, patient group studied, study type, relevant outcomes and results of these papers are tabulated. The papers identified included 683 adult patients with bronchogenic cysts. There was a substantial variation between the papers in the proportion of patients presenting with symptoms (6–79%), and all patients with symptoms were managed surgically. The majority of asymptomatic patients underwent empirical surgery to prevent the development of symptoms, to confirm the diagnosis and to rule out malignant transformation. A total of 74 asymptomatic patients were treated conservatively or had definitive diagnosis or treatment delayed. The longest period of observation was 22 years. In total, 33 (45%) of asymptomatic patients eventually developed symptoms requiring surgery. There was no evidence to suggest that surgery following a cyst-related complication increased the postoperative morbidity or mortality, although it was noted to increase the technical difficulty of the procedure. There were no descriptions of misdiagnosis of malignancy as bronchogenic cyst, but 5 (0.7%) of the 683 cysts studied were found to be associated with malignant cells in the cyst wall. The figures cited, however, represent only symptomatic or incidental presentations. As the prevalence of these otherwise benign entities is not known, the rates of progression to symptoms and associated malignancy may be lower than those described. We would advocate informing asymptomatic patients diagnosed with bronchogenic cyst of the 20% morbidity of surgery whether immediate or delayed, the 45% risk of developing symptoms, some of which may be serious, and the 0.7% risk of malignancy. Should patients opt for conservative management, this can be offered only if close long-term follow-up can be guaranteed.
1. Introduction
A best evidence topic was constructed according to a structured protocol. This is fully described in the ICVTS [1].
2. Three-part question
In [adult patients with asymptomatic bronchogenic cysts] does [surgery] improve [symptom-free survival]?
3. Clinical scenario
An otherwise well patient is found to have a well-circumscribed lesion on routine chest X-ray and a computed tomography (CT)-scan is arranged. The report states that there are characteristic features of a bronchogenic cyst and you decide to discuss the management with your colleagues. You suggest that as the patient is asymptomatic, the patient could potentially be treated conservatively with regular follow-up, but others feel that excision is appropriate. Opinion appears to be divided in the department so you seek evidence to determine best practice.
4. Search strategy
Medline 1950 to May 2010 using PubMed interface.
‘Bronchogenic Cyst’[Mesh] AND (‘Surgical Procedures, Operative’[Mesh] OR ‘surgery’[Subheading] OR ‘Thoracic Surgery, Video-Assisted’[Mesh] OR ‘Thoracic Surgery’[Mesh] OR ‘Video-Assisted Surgery’[Mesh]).
The search was limited to human studies involving adults.
5. Search outcome
Three hundred and ten papers were found using the reported search. From these 23 papers were identified that provided the best evidence to answer the question. These are presented in Table 1 .
Author, date and country | Patient group | Outcomes | Key results | Comments |
Study type | ||||
(level of evidence) | ||||
Laberge et al., | Review of literature | Presenting complaint | Dysphagia, dyspnoea, | Highly heterogenous |
Semin Pediatr Surg, | regarding asymptomatic | infection, haemoptysis | group including all adult | |
2005, Canada, [2] | congenital lung | and haemothorax (no | and paediatric congenital | |
malformations, | figures of incidence) | lung malformations | ||
Review (2a) | including bronchogenic | |||
cysts, in a paediatric | Postoperative | Adult postoperative | Two cases of | |
population | complications | complications not | malignancy in | |
discussed | bronchogenic cysts | |||
Includes papers on adult | noted in adult patients | |||
congenital lung | Complications of | One adult patient with | ||
malformations | conservative | abnormal chest X-ray | Conclusions based on | |
management | eventually presented | management of | ||
10 years later with | paediatric malformations | |||
dyspnoea and was found | but recommend early | |||
to have | excision of bronchogenic | |||
bronchioloalveolar | cysts due to progression | |||
carcinoma associated | to symptoms, malignant | |||
with bronchogenic cyst. | potential and | |||
Another patient had ‘long | complications of | |||
standing’ history of cyst | symptomatic cysts | |||
infections and later found | ||||
to have associated | ||||
mesenchymal malignancy | ||||
Sarper et al., Tex | 22 patients from one | Presenting complaint | 45% presented with | Recommended surgical |
Heart Inst J, 2003, | centre, over 15 years | severe haemoptysis, | resection of all suspected | |
Turkey, [3] | pneumothorax and | bronchogenic cysts in | ||
5.2-year median | pleuritis, oesophageal | operable candidates due | ||
Retrospective | postoperative follow-up | compression, infected | to difficulties in | |
cohort study (2b) | cyst, or postobstructive | establishing definitive | ||
pneumonia. | diagnoses and frequency | |||
82% presented with | of complications | |||
symptoms of cough, | ||||
pain, dyspnoea, | ||||
dysphagia or infection | ||||
Postoperative | 5% (one patient) | |||
complications | persistent air leak | |||
No recurrence | ||||
No late sequelae | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Kanemitsu et al., | 17 patients (16 adults | Presenting complaint | 29% symptomatic (9% | It is noted that the |
Surg Today, 1999, | and one paediatric) from | of mediastinal cysts, 67% | advanced age of some | |
Japan, [4] | one centre, over | of intrapulmonary cysts) | patients at presentation | |
30 years | with cough, sputum, | suggests that these cysts | ||
Retrospective | fever, pain or weight loss | can remain | ||
cohort study (2b) | 30-month median | asymptomatic forever. | ||
postoperative follow-up | Preoperative | 69% diagnosed with CT | The authors state that the | |
diagnosis | 100% diagnosed with | most appropriate | ||
MRI | treatment for | |||
asymptomatic cysts is | ||||
Operative findings | 41% adhesions, 6% | controversial but due to | ||
complicated | diagnostic limitations and | |||
the possibility of | ||||
Postoperative | No postoperative | symptoms arising or | ||
complications | complications. | malignant transformation | ||
No recurrence | that surgery is advocated | |||
Complications of | None in one patient | |||
incomplete resection | where residual tissue | |||
ablated with | ||||
electro-cautery | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Cioffi et al., Chest, | 27 adults of whom | Presenting complaint | 50% chest pain, 13% | Conclude that all patients |
1998, Italy, [5] | 16 with bronchogenic | cough, 6% epigastric | should have surgical | |
cyst from one centre, | pain. | resection for definitive | ||
Retrospective | over 20 years | 44% asymptomatic | diagnosis and to | |
cohort study (2b) | (incidental finding) | minimise complications | ||
Four-year median | that might arise from | |||
postoperative follow-up | Preoperative | 100% preoperatively | symptomatic cysts | |
diagnosis | diagnosed with CT, EUS | |||
Postoperative | No postoperative | |||
complications | complications | |||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Aktogu et al., Eur | 31 patients (30 adults | Presenting complaint | 19% superior vena cava | Thirteen patients |
Respir J, 1996, | and one paediatric) from | syndrome, tracheal | symptomatic at the time | |
Turkey, [6] | one centre, over | compression, | of surgery had complex | |
19 years | pneumothorax, pleurisy | peri-cystic adhesions or | ||
Retrospective | or pneumonia. | fistulisation. | ||
cohort study (2b) | Follow-up 2–10 years | 81% cough, infection, | Surgical treatment of | |
postoperatively | pain, dyspnoea, anorexia/ | asymptomatic cysts is | ||
weight loss, haemoptysis | recommended to avoid | |||
potentially | ||||
Preoperative | 19% asymptomatic | life-threatening | ||
diagnosis | (incidental finding). | complications and for | ||
71% undiagnosed with | definitive diagnosis | |||
CT | ||||
Postoperative | No recurrence | |||
complications | ||||
Complications of | Two of six initially | |||
conservative | asymptomatic patients | |||
management | either had cyst | |||
enlargement or became | ||||
symptomatic | ||||
Ribet et al., Ann | 41 patients from one | Presenting complaint | 80% symptomatic | Uncertain what |
Thorac Surg, 1996, | centre, including 20 | (cough, pain, purulent | proportion of | |
France, [7] | paediatric cases, over | sputum, haemoptysis and | bronchogenic cysts | |
25 years | dyspnoea) in adult life | remain asymptomatic | ||
Retrospective | and long-term prognosis | |||
cohort study (2b) | 2.6-year mean | Preoperative | 45% undiagnosed | unpredictable. |
postoperative follow-up | diagnosis | Preventative surgery | ||
recommended | ||||
Postoperative | 5% (one patient) | |||
complications | bronchial fistula. | |||
No recurrence in 88% | ||||
(remainder lost to | ||||
follow-up). | ||||
No deaths | ||||
Complications of | 15% (three patients) | |||
conservative | followed for 11 months, | |||
management | five years and unknown | |||
length of time since | ||||
diagnosis developed no | ||||
symptoms but were | ||||
operated on. | ||||
15% (three patients) | ||||
initially with cough, | ||||
recurrent bronchitis and | ||||
no symptoms | ||||
(respectively) refused | ||||
surgery but lost to | ||||
follow-up | ||||
Cuypers et al., Eur | 20 adult patients from | Presenting complaint | 30% symptomatic | The authors found the |
J Cardiothorac | one centre, over | (pneumonia, abscess, | risk of malignancy and | |
Surg, 1996, | 18 years | dysphagia and cough) | cyst-related | |
Belgium, [8] | 70% asymptomatic | complications to be | ||
No long-term follow-up | justification for operative | |||
Retrospective | Preoperative | 25% undiagnosed after | treatment in all cases of | |
cohort study (2b) | diagnosis | CT, bronchoscopy, | bronchogenic cysts | |
barium swallow or echo | ||||
(all cases had CT with | ||||
dense fluid) | ||||
Postoperative | No postoperative | |||
complications | morbidity; one case | |||
histologically associated | ||||
with squamous cell | ||||
carcinoma | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Ribet et al., J | 69 patients from one | Presenting complaint | 63.7% symptomatic | Due to the frequency of |
Thorac Cardiovasc | centre, including 24 | pain, respiratory tract | late complications with | |
Surg, 1995, France, | paediatric cases, over | infection, cough, | asymptomatic cysts and | |
[9] | 25 years | dyspnoea, dysphagia, | the unpredictable | |
heartburn) | prognosis, preventative | |||
Retrospective | 4.2-year mean | surgery was | ||
cohort study (2b) | postoperative follow-up | Preoperative | 22% initially | recommended |
diagnosis | misdiagnosed (11% not | |||
as bronchogenic cyst; | ||||
11% as bronchogenic | ||||
cyst when actually cystic | ||||
neurogenic tumour, | ||||
benign lymphoma and | ||||
haemolymphangioma) | ||||
Postoperative | 13.4% postoperative | |||
complications | morbidity (infection, | |||
chylothorax and phrenic | ||||
paresis). | ||||
12% symptoms of pain | ||||
or dyspnoea | ||||
postoperative (7% | ||||
symptomatic | ||||
preoperative, 5% | ||||
asymptomatic) | ||||
Complications of | 11% refused operation, | |||
conservative | only 4% followed up | |||
management | (two patients): one | |||
remained asymptomatic, | ||||
one died of generalised | ||||
malignancy of unknown | ||||
origin. | ||||
7% had incomplete | ||||
operations but cyst | ||||
remained stable or had no | ||||
recurrence | ||||
Patel et al., Chest, | 18 adult patients from | Presenting complaint | 44% symptomatic | There was no statistical |
1994, USA, [10] | one centre, over | (cough, pain) of which | difference in the | |
19-year period | 11% serious (dyspnea, | frequency of | ||
Retrospective | infection). | intraoperative difficulties | ||
cohort study (2b) | Follow-up 12 | 56% asymptomatic | or postoperative | |
months to 11 years | complications between | |||
(mean not given) | Preoperative | 37.5% undiagnosed with | asymptomatic and | |
diagnosis | CT, angio, USS, FNA, | symptomatic patients. | ||
barium swallow, | The authors concluded, | |||
bronchoscopy or | however, that surgery is | |||
mediastinoscopy | advocated in | |||
Operative difficulties | No difference in | asymptomatic cysts due | ||
frequency of operative | to the potential for | |||
difficulties between | complications, incorrect | |||
symptomatic and | diagnosis or progression | |||
asymptomatic patients | to symptoms | |||
(P=0.0656) | ||||
Postoperative | No difference (P=0.596) | |||
complications | in complication rate | |||
between preoperatively | ||||
asymptomatic (14%) or | ||||
symptomatic (27%) | ||||
patients. Complications | ||||
included phrenic nerve | ||||
paresis. | ||||
11% delayed | ||||
complications | ||||
(oesophageal stricture | ||||
and recurrence) | ||||
Complications of | Three out of seven | |||
conservative | asymptomatic patients | |||
management | followed up developed | |||
symptoms; four patients | ||||
lost to follow-up | ||||
Suen et al., Ann | 42 patients from one | Presenting complaint | 50% symptomatic (pain, | Complete excision |
Thorac Surg, | centre, over 30 years | cough, fever, dysphagia, | recommended in most | |
1993, USA, [11] | purulent sputum, | cases to relieve | ||
haemoptysis, dyspnoea). | symptoms, prevent | |||
Retrospective | 26% complicated | complications and | ||
cohort study (2b) | (dysphagia, haemorrhage, | confirm diagnosis | ||
infection and one patient | ||||
with adenocarcinoma in | ||||
cyst) | ||||
Preoperative | 59% no preoperative | |||
diagnosis | diagnosis (more recent | |||
cases, better success | ||||
rate) | ||||
Postoperative | 5% postoperative | |||
complications | complications (wound | |||
infection and C. Diff | ||||
colitis). | ||||
No recurrence. | ||||
No deaths | ||||
Complications of | Two patients treated | |||
conservative | conservatively (one | |||
management | follow-up only and one | |||
drainage) with no | ||||
complications | ||||
St-Georges et al., | 86 patients from one | Presenting complaint | 72% patients | 35/86 (41%) of patients |
Ann Thorac Surg, | centre, over 20 years | symptomatic by time of | operated on had major | |
1991, Canada, [12] | surgery (57% | operative difficulties: all | ||
progressive symptoms | these were patients with | |||
Retrospective | and 15% acute). | symptoms | ||
cohort study (2b) | 53% patients with >1 | preoperatively | ||
symptom (chest pain, | ||||
cough, dyspnoea, fever, | Resection recommended | |||
sputum, anorexia/weight | as majority of patients | |||
loss, dysphagia, | eventually develop | |||
haemoptysis). | symptoms or | |||
38% with complications | complications | |||
(fistula with airway, | ||||
ulceration cyst wall, | ||||
haemorrhage, infection, | ||||
bronchial atresia) | ||||
Preoperative | 57% patients presumed | |||
diagnosis | diagnosis at surgery after | |||
CT and angiography. | ||||
Positive diagnosis never | ||||
made preoperatively | ||||
Operative findings | 41% had complicated | |||
cysts (fistula, ulcer, | ||||
haemorrhage, infection, | ||||
atresia) at operation | ||||
Postoperative | 11% intraoperative | |||
complications | complications (vagal | |||
trunk division, segmental | ||||
bronchus laceration, | ||||
oesophageal mucosal | ||||
laceration). | ||||
9% postoperative | ||||
complications | ||||
(atelectasis, pleural | ||||
effusion, wound | ||||
infection, transient | ||||
Horner's syndrome, | ||||
respiratory failure | ||||
requiring tracheostomy | ||||
and haemothorax) with | ||||
major postoperative | ||||
complications in | ||||
symptomatic patients | ||||
Complications of | 37 patients were | |||
conservative | followed up | |||
management | conservatively, and | |||
13 (35%) were still | ||||
asymptomatic at time of | ||||
operation | ||||
Cartmill and | 20 patients from one | Presenting complaint | 75% symptomatic (chest | Advocate surgery for |
Hughes, Aust N Z J | centre, over 10 years. | pain, cough, | symptom relief, | |
Surg, 1989, | Included some | haemoptysis, dysphagia). | exclusion of malignancy | |
Australia, [13] | paediatric patients (not | 5% serious complications | and prevention of | |
known how many) | catastrophic | |||
Retrospective | Preoperative | 45% diagnosed with CT, | complications | |
cohort study (2b) | Mean postoperative | diagnosis | barium swallow and | |
follow-up 67 months | aortography | |||
Postoperative | 5% (one patient) multiple | |||
complications | PE. | |||
No recurrence. | ||||
No death | ||||
Complications of | 5% (one patient) declined | |||
conservative | surgery initially but | |||
management | returned for elective | |||
treatment after six years. | ||||
Reason not given | ||||
Coselli et al., Ann | Eight patients from one | Presenting complaint | 75% symptomatic | Recommend excision to |
Thorac Surg, 1987, | centre, over 11 years | (dysphagia, epigastric | establish diagnosis, | |
USA, [14] | pain, respiratory distress, | alleviate symptoms and | ||
dyspnoea, chest pain) | prevent complications | |||
Case series (4) | ||||
Preoperative | With use of CT; operated | |||
diagnosis | on to confirm diagnosis | |||
Operative findings | Previously infected cysts | |||
more difficult and | ||||
hazardous to excise | ||||
Ge et al., Chin Med | 22 patients from one | Presenting complaint | 91% symptomatic (chest | Recommend that |
Sci J, 1995, China | centre, over 20 years. | pain, dyspnoea, cough, | asymptomatic | |
[15] | Included paediatric | fever, infection, | bronchogenic cysts | |
patients, but not | dysphagia, haemoptysis) | should be excised | ||
Retrospective | specified how many | because of the high risk | ||
cohort study (2b) | Preoperative | 36.4% diagnosed with | of complications, | |
Mean postoperative | diagnosis | CT | although this conclusion | |
follow-up seven years | was not tested in the | |||
Postoperative | No recurrence. | study | ||
complications | No complications. | |||
No mortality | ||||
Takeda et al., | 105 patients (with any | Presenting complaint | 40% symptomatic (chest | Heterogenous population. |
Chest, 2003, USA | mediastinal cyst) from | pain, dyspnoea, cough, | Data extracted for | |
[16] | one centre including | fever, sputum, | bronchogenic cysts only. | |
45 adults and six | dysphagia, haemoptysis) | Three patients had | ||
Retropective cohort | paediatric patients with | complicated surgery due | ||
study (2b) | bronchogenic cysts, | Preoperative | Could not extract data | to peri-cystic adhesions. |
over 50-year period | diagnosis | but improved diagnostic | Preventative resection | |
capabilities of MRI | preferred because of | |||
acknowledged | unpredictable clinical | |||
behaviour | ||||
Postoperative | Could not extract data | |||
complications | but described as | |||
‘acceptable’ by authors | ||||
Complications of | Two patients refused | |||
conservative | treatment: outcome not | |||
management | described | |||
Gursoy et al., Saudi | 28 adult patients with | Presenting complaint | 71% symptomatic | Six additional patients |
Med J, 2009, | preoperative diagnosis | (dyspnoea, chest pain, | excluded as preoperative | |
Turkey, [17] | of bronchogenic cyst | cough, fever) | diagnosis of | |
from one centre over | bronchogenic cyst not | |||
Retrospective | seven years | Preoperative | 82.4% correctly | confirmed histologically |
cohort study (2b) | diagnosis | diagnosed with CT | ||
36-month mean | Surgical resection | |||
postoperative | Postoperative | 11% early complication | recommended in | |
follow-up | complications | rate (wound infection, | asymptomatic patients | |
prolonged air leak, | because of the possibility | |||
pneumoperitoneum). | of malignant | |||
7% late complication | transformation and | |||
(dyspnea, | anatomic complications | |||
pneumothorax). | of delayed surgery | |||
No mortality | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Liu et al., Chin Med | 50 adult patients with | Presenting complaint | 66% symptomatic | Recommend surgical |
Sci J, 2009, China, | histopathologically | (cough, chest pain, | resection to confirm | |
[18] | proven bronchogenic | hemoptysis, dyspnoea, | diagnosis, avoid | |
cyst from one centre of | fever, dysphagia, | development of | ||
Retrospective | 24 years | paralysis, hoarseness). | symptoms or malignant | |
cohort study (2b) | 26% serious | change. Conclusions | ||
6.5-year mean | complications | drawn upon findings of | ||
follow-up | other studies | |||
Preoperative | 40% diagnosed | |||
diagnosis | preoperatively. | |||
14% misdiagnosed after | ||||
all investigations | ||||
Postoperative | 4% early complication | |||
complications | (persistent air leak, | |||
hoarseness). | ||||
No late complications. | ||||
No mortality | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Kosar et al., Heart | 29 patients from one | Presenting complaint | 86% symptomatic | The authors suggest |
Lung Circ, 2009 | centre, including 13 | (cough, sputum, pain, | there is a ‘general | |
Turkey, [19] | paediatric patients, | breathlessness, | consensus’ that all | |
over 15 years treated | haemoptysis, fever) | bronchogenic cysts | ||
Retrospective | with either resection | should be operated on to | ||
cohort study (2b) | or de-epithelialisation | Preoperative | 89.7% diagnosed | avoid development of |
diagnosis | preoperatively using | symptoms or | ||
predominantly CT | complications | |||
Postoperative | 17% early complication | |||
complications | rate (pneumonia, wound | |||
infections, prolonged air | ||||
leak); higher in | ||||
complicated cysts. | ||||
No late complications. | ||||
No mortality. | ||||
Recurrence in | ||||
de-epithelialised group | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Limaïauiem et al., | 33 patients from one | Presenting complaint | 94% symptomatic (chest | Management of all |
Lung, 2008, | centre over six years | pain, cough, | bronchogenic cysts | |
Tunisia, [20] | haemoptysis, dyspnoea, | based on complete | ||
Follow-up between | fever, dysphagia) | surgical excision. | ||
Retrospective | one and 51 months | Conclude that definitive | ||
cohort study (2b) | Preoperative | Correct diagnosis in | diagnosis is by histology | |
diagnosis | 33.3% | only and that complete | ||
surgical excision is | ||||
Postoperative | 14% Early complications | mandatory, although | ||
complications | (pneumothorax, | conclusions based on | ||
haemorrhage, pleural | findings of other studies | |||
effusion, seizure). | ||||
No late complications. | ||||
No mortality | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Granato et al., | 30 adult patients treated | Presenting complaint | 30% symptomatic | Two symptomatic cysts |
Asian Cardiovasc | for bronchogenic cysts | (cough, sputum, pain, | complicated | |
Thorac Ann, 2009, | from one centre, over | fever, weakness) | intraoperatively by severe | |
Italy, [21] | 32 years | adhesions | ||
Preoperative | Correct diagnosis in | |||
Retrospective | diagnosis | 100% (CT or CT and | One case of large-cell | |
cohort study (2b) | MRI) | anaplastic carcinoma in | ||
wall of cyst | ||||
Postoperative | 10% intraoperative | |||
complications | complications. | Excision of | ||
10% postoperative | asymptomatic cysts | |||
complications | advocated to avoid | |||
complex surgery and | ||||
Complications of | No patients | complications, and also | ||
conservative | conservatively managed | to reduce malignant | ||
management | potential | |||
De Giacomo et al., | 30 adult patients from | Presenting complaint | 37% symptomatic | Authors feel that patients |
Eur J Cardiothorac Surg | one centre over | (cough, pain, dysphagia) | cannot be completely | |
2009, Italy, [22] | 12 years | assured about | ||
Postoperative | None | conservative | ||
Retrospective | Follow-up | complications | management but | |
cohort study (2b) | 3–120 months | acknowledge the | ||
Complications of | Asymptomatic patients | management is | ||
conservative | requested surgery | controversial | ||
management | because of enlarging | |||
cysts, risk of | ||||
complication or fear of | ||||
malignancy | ||||
Costa Júnior Ada | 60 patients with | Presenting complaint | 92% symptomatic | Heterogenous group of |
et al., J Bras | pulmonary | (cough, dyspnoea, pain, | patients and disorders. | |
Pneumol, 2008, | malformations | infection) | Difficult to extract | |
Brazil, [23] | (including 27 with | bronchogenic cyst data | ||
bronchogenic cyst) | Preoperative | ‘Frequent’ misdiagnosis | ||
Retrospective | from one centre | diagnosis | Prognosis noted to be | |
cohort study (2b) | over 35 years, | unpredictable. One | ||
including 40 paediatric | Postoperative | 23% (pneumonia, | patient found to have | |
patients | complications | atelectasis, empyema, | adenocarcinoma in wall | |
sepsis). | of cyst | |||
3.3% mortality | ||||
Complications of | Diagnosis/treatment | |||
conservative | delayed in three patients | |||
management | up to 36 months (mean | |||
15): outcome in these | ||||
patients not specified | ||||
Weber et al., | 12 patients from a | Presenting complaint | 42% symptomatic | Agree that management |
Ann Thorac Surg, | single centre | (cough, pain, pneumonia) | of asymptomatic cysts is | |
2004, Switzerland, | undergoing | controversial but that | ||
[24] | video-assisted | Preoperative | 100% correct diagnosis | there appears to be no |
thoracoscopic surgery | diagnosis | with CT with or without | need for urgent surgery | |
Retrospective | for bronchogenic | MRI. MRI noted to be | in these cases provided | |
cohort study (2b) | cysts, over seven years | superior | that a simple cyst has | |
been clearly diagnosed | ||||
40.5-month mean | Postoperative | None | ||
follow-up | complications | |||
Complications of | Six patients observed for | |||
conservative | between two and | |||
management | 22 years without | |||
complications.Three | ||||
developed mild | ||||
symptoms. | ||||
Three patients eventually | ||||
requested surgery | ||||
because of fear of | ||||
malignancy/complications | ||||
or enlarging cyst |
Author, date and country | Patient group | Outcomes | Key results | Comments |
Study type | ||||
(level of evidence) | ||||
Laberge et al., | Review of literature | Presenting complaint | Dysphagia, dyspnoea, | Highly heterogenous |
Semin Pediatr Surg, | regarding asymptomatic | infection, haemoptysis | group including all adult | |
2005, Canada, [2] | congenital lung | and haemothorax (no | and paediatric congenital | |
malformations, | figures of incidence) | lung malformations | ||
Review (2a) | including bronchogenic | |||
cysts, in a paediatric | Postoperative | Adult postoperative | Two cases of | |
population | complications | complications not | malignancy in | |
discussed | bronchogenic cysts | |||
Includes papers on adult | noted in adult patients | |||
congenital lung | Complications of | One adult patient with | ||
malformations | conservative | abnormal chest X-ray | Conclusions based on | |
management | eventually presented | management of | ||
10 years later with | paediatric malformations | |||
dyspnoea and was found | but recommend early | |||
to have | excision of bronchogenic | |||
bronchioloalveolar | cysts due to progression | |||
carcinoma associated | to symptoms, malignant | |||
with bronchogenic cyst. | potential and | |||
Another patient had ‘long | complications of | |||
standing’ history of cyst | symptomatic cysts | |||
infections and later found | ||||
to have associated | ||||
mesenchymal malignancy | ||||
Sarper et al., Tex | 22 patients from one | Presenting complaint | 45% presented with | Recommended surgical |
Heart Inst J, 2003, | centre, over 15 years | severe haemoptysis, | resection of all suspected | |
Turkey, [3] | pneumothorax and | bronchogenic cysts in | ||
5.2-year median | pleuritis, oesophageal | operable candidates due | ||
Retrospective | postoperative follow-up | compression, infected | to difficulties in | |
cohort study (2b) | cyst, or postobstructive | establishing definitive | ||
pneumonia. | diagnoses and frequency | |||
82% presented with | of complications | |||
symptoms of cough, | ||||
pain, dyspnoea, | ||||
dysphagia or infection | ||||
Postoperative | 5% (one patient) | |||
complications | persistent air leak | |||
No recurrence | ||||
No late sequelae | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Kanemitsu et al., | 17 patients (16 adults | Presenting complaint | 29% symptomatic (9% | It is noted that the |
Surg Today, 1999, | and one paediatric) from | of mediastinal cysts, 67% | advanced age of some | |
Japan, [4] | one centre, over | of intrapulmonary cysts) | patients at presentation | |
30 years | with cough, sputum, | suggests that these cysts | ||
Retrospective | fever, pain or weight loss | can remain | ||
cohort study (2b) | 30-month median | asymptomatic forever. | ||
postoperative follow-up | Preoperative | 69% diagnosed with CT | The authors state that the | |
diagnosis | 100% diagnosed with | most appropriate | ||
MRI | treatment for | |||
asymptomatic cysts is | ||||
Operative findings | 41% adhesions, 6% | controversial but due to | ||
complicated | diagnostic limitations and | |||
the possibility of | ||||
Postoperative | No postoperative | symptoms arising or | ||
complications | complications. | malignant transformation | ||
No recurrence | that surgery is advocated | |||
Complications of | None in one patient | |||
incomplete resection | where residual tissue | |||
ablated with | ||||
electro-cautery | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Cioffi et al., Chest, | 27 adults of whom | Presenting complaint | 50% chest pain, 13% | Conclude that all patients |
1998, Italy, [5] | 16 with bronchogenic | cough, 6% epigastric | should have surgical | |
cyst from one centre, | pain. | resection for definitive | ||
Retrospective | over 20 years | 44% asymptomatic | diagnosis and to | |
cohort study (2b) | (incidental finding) | minimise complications | ||
Four-year median | that might arise from | |||
postoperative follow-up | Preoperative | 100% preoperatively | symptomatic cysts | |
diagnosis | diagnosed with CT, EUS | |||
Postoperative | No postoperative | |||
complications | complications | |||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Aktogu et al., Eur | 31 patients (30 adults | Presenting complaint | 19% superior vena cava | Thirteen patients |
Respir J, 1996, | and one paediatric) from | syndrome, tracheal | symptomatic at the time | |
Turkey, [6] | one centre, over | compression, | of surgery had complex | |
19 years | pneumothorax, pleurisy | peri-cystic adhesions or | ||
Retrospective | or pneumonia. | fistulisation. | ||
cohort study (2b) | Follow-up 2–10 years | 81% cough, infection, | Surgical treatment of | |
postoperatively | pain, dyspnoea, anorexia/ | asymptomatic cysts is | ||
weight loss, haemoptysis | recommended to avoid | |||
potentially | ||||
Preoperative | 19% asymptomatic | life-threatening | ||
diagnosis | (incidental finding). | complications and for | ||
71% undiagnosed with | definitive diagnosis | |||
CT | ||||
Postoperative | No recurrence | |||
complications | ||||
Complications of | Two of six initially | |||
conservative | asymptomatic patients | |||
management | either had cyst | |||
enlargement or became | ||||
symptomatic | ||||
Ribet et al., Ann | 41 patients from one | Presenting complaint | 80% symptomatic | Uncertain what |
Thorac Surg, 1996, | centre, including 20 | (cough, pain, purulent | proportion of | |
France, [7] | paediatric cases, over | sputum, haemoptysis and | bronchogenic cysts | |
25 years | dyspnoea) in adult life | remain asymptomatic | ||
Retrospective | and long-term prognosis | |||
cohort study (2b) | 2.6-year mean | Preoperative | 45% undiagnosed | unpredictable. |
postoperative follow-up | diagnosis | Preventative surgery | ||
recommended | ||||
Postoperative | 5% (one patient) | |||
complications | bronchial fistula. | |||
No recurrence in 88% | ||||
(remainder lost to | ||||
follow-up). | ||||
No deaths | ||||
Complications of | 15% (three patients) | |||
conservative | followed for 11 months, | |||
management | five years and unknown | |||
length of time since | ||||
diagnosis developed no | ||||
symptoms but were | ||||
operated on. | ||||
15% (three patients) | ||||
initially with cough, | ||||
recurrent bronchitis and | ||||
no symptoms | ||||
(respectively) refused | ||||
surgery but lost to | ||||
follow-up | ||||
Cuypers et al., Eur | 20 adult patients from | Presenting complaint | 30% symptomatic | The authors found the |
J Cardiothorac | one centre, over | (pneumonia, abscess, | risk of malignancy and | |
Surg, 1996, | 18 years | dysphagia and cough) | cyst-related | |
Belgium, [8] | 70% asymptomatic | complications to be | ||
No long-term follow-up | justification for operative | |||
Retrospective | Preoperative | 25% undiagnosed after | treatment in all cases of | |
cohort study (2b) | diagnosis | CT, bronchoscopy, | bronchogenic cysts | |
barium swallow or echo | ||||
(all cases had CT with | ||||
dense fluid) | ||||
Postoperative | No postoperative | |||
complications | morbidity; one case | |||
histologically associated | ||||
with squamous cell | ||||
carcinoma | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Ribet et al., J | 69 patients from one | Presenting complaint | 63.7% symptomatic | Due to the frequency of |
Thorac Cardiovasc | centre, including 24 | pain, respiratory tract | late complications with | |
Surg, 1995, France, | paediatric cases, over | infection, cough, | asymptomatic cysts and | |
[9] | 25 years | dyspnoea, dysphagia, | the unpredictable | |
heartburn) | prognosis, preventative | |||
Retrospective | 4.2-year mean | surgery was | ||
cohort study (2b) | postoperative follow-up | Preoperative | 22% initially | recommended |
diagnosis | misdiagnosed (11% not | |||
as bronchogenic cyst; | ||||
11% as bronchogenic | ||||
cyst when actually cystic | ||||
neurogenic tumour, | ||||
benign lymphoma and | ||||
haemolymphangioma) | ||||
Postoperative | 13.4% postoperative | |||
complications | morbidity (infection, | |||
chylothorax and phrenic | ||||
paresis). | ||||
12% symptoms of pain | ||||
or dyspnoea | ||||
postoperative (7% | ||||
symptomatic | ||||
preoperative, 5% | ||||
asymptomatic) | ||||
Complications of | 11% refused operation, | |||
conservative | only 4% followed up | |||
management | (two patients): one | |||
remained asymptomatic, | ||||
one died of generalised | ||||
malignancy of unknown | ||||
origin. | ||||
7% had incomplete | ||||
operations but cyst | ||||
remained stable or had no | ||||
recurrence | ||||
Patel et al., Chest, | 18 adult patients from | Presenting complaint | 44% symptomatic | There was no statistical |
1994, USA, [10] | one centre, over | (cough, pain) of which | difference in the | |
19-year period | 11% serious (dyspnea, | frequency of | ||
Retrospective | infection). | intraoperative difficulties | ||
cohort study (2b) | Follow-up 12 | 56% asymptomatic | or postoperative | |
months to 11 years | complications between | |||
(mean not given) | Preoperative | 37.5% undiagnosed with | asymptomatic and | |
diagnosis | CT, angio, USS, FNA, | symptomatic patients. | ||
barium swallow, | The authors concluded, | |||
bronchoscopy or | however, that surgery is | |||
mediastinoscopy | advocated in | |||
Operative difficulties | No difference in | asymptomatic cysts due | ||
frequency of operative | to the potential for | |||
difficulties between | complications, incorrect | |||
symptomatic and | diagnosis or progression | |||
asymptomatic patients | to symptoms | |||
(P=0.0656) | ||||
Postoperative | No difference (P=0.596) | |||
complications | in complication rate | |||
between preoperatively | ||||
asymptomatic (14%) or | ||||
symptomatic (27%) | ||||
patients. Complications | ||||
included phrenic nerve | ||||
paresis. | ||||
11% delayed | ||||
complications | ||||
(oesophageal stricture | ||||
and recurrence) | ||||
Complications of | Three out of seven | |||
conservative | asymptomatic patients | |||
management | followed up developed | |||
symptoms; four patients | ||||
lost to follow-up | ||||
Suen et al., Ann | 42 patients from one | Presenting complaint | 50% symptomatic (pain, | Complete excision |
Thorac Surg, | centre, over 30 years | cough, fever, dysphagia, | recommended in most | |
1993, USA, [11] | purulent sputum, | cases to relieve | ||
haemoptysis, dyspnoea). | symptoms, prevent | |||
Retrospective | 26% complicated | complications and | ||
cohort study (2b) | (dysphagia, haemorrhage, | confirm diagnosis | ||
infection and one patient | ||||
with adenocarcinoma in | ||||
cyst) | ||||
Preoperative | 59% no preoperative | |||
diagnosis | diagnosis (more recent | |||
cases, better success | ||||
rate) | ||||
Postoperative | 5% postoperative | |||
complications | complications (wound | |||
infection and C. Diff | ||||
colitis). | ||||
No recurrence. | ||||
No deaths | ||||
Complications of | Two patients treated | |||
conservative | conservatively (one | |||
management | follow-up only and one | |||
drainage) with no | ||||
complications | ||||
St-Georges et al., | 86 patients from one | Presenting complaint | 72% patients | 35/86 (41%) of patients |
Ann Thorac Surg, | centre, over 20 years | symptomatic by time of | operated on had major | |
1991, Canada, [12] | surgery (57% | operative difficulties: all | ||
progressive symptoms | these were patients with | |||
Retrospective | and 15% acute). | symptoms | ||
cohort study (2b) | 53% patients with >1 | preoperatively | ||
symptom (chest pain, | ||||
cough, dyspnoea, fever, | Resection recommended | |||
sputum, anorexia/weight | as majority of patients | |||
loss, dysphagia, | eventually develop | |||
haemoptysis). | symptoms or | |||
38% with complications | complications | |||
(fistula with airway, | ||||
ulceration cyst wall, | ||||
haemorrhage, infection, | ||||
bronchial atresia) | ||||
Preoperative | 57% patients presumed | |||
diagnosis | diagnosis at surgery after | |||
CT and angiography. | ||||
Positive diagnosis never | ||||
made preoperatively | ||||
Operative findings | 41% had complicated | |||
cysts (fistula, ulcer, | ||||
haemorrhage, infection, | ||||
atresia) at operation | ||||
Postoperative | 11% intraoperative | |||
complications | complications (vagal | |||
trunk division, segmental | ||||
bronchus laceration, | ||||
oesophageal mucosal | ||||
laceration). | ||||
9% postoperative | ||||
complications | ||||
(atelectasis, pleural | ||||
effusion, wound | ||||
infection, transient | ||||
Horner's syndrome, | ||||
respiratory failure | ||||
requiring tracheostomy | ||||
and haemothorax) with | ||||
major postoperative | ||||
complications in | ||||
symptomatic patients | ||||
Complications of | 37 patients were | |||
conservative | followed up | |||
management | conservatively, and | |||
13 (35%) were still | ||||
asymptomatic at time of | ||||
operation | ||||
Cartmill and | 20 patients from one | Presenting complaint | 75% symptomatic (chest | Advocate surgery for |
Hughes, Aust N Z J | centre, over 10 years. | pain, cough, | symptom relief, | |
Surg, 1989, | Included some | haemoptysis, dysphagia). | exclusion of malignancy | |
Australia, [13] | paediatric patients (not | 5% serious complications | and prevention of | |
known how many) | catastrophic | |||
Retrospective | Preoperative | 45% diagnosed with CT, | complications | |
cohort study (2b) | Mean postoperative | diagnosis | barium swallow and | |
follow-up 67 months | aortography | |||
Postoperative | 5% (one patient) multiple | |||
complications | PE. | |||
No recurrence. | ||||
No death | ||||
Complications of | 5% (one patient) declined | |||
conservative | surgery initially but | |||
management | returned for elective | |||
treatment after six years. | ||||
Reason not given | ||||
Coselli et al., Ann | Eight patients from one | Presenting complaint | 75% symptomatic | Recommend excision to |
Thorac Surg, 1987, | centre, over 11 years | (dysphagia, epigastric | establish diagnosis, | |
USA, [14] | pain, respiratory distress, | alleviate symptoms and | ||
dyspnoea, chest pain) | prevent complications | |||
Case series (4) | ||||
Preoperative | With use of CT; operated | |||
diagnosis | on to confirm diagnosis | |||
Operative findings | Previously infected cysts | |||
more difficult and | ||||
hazardous to excise | ||||
Ge et al., Chin Med | 22 patients from one | Presenting complaint | 91% symptomatic (chest | Recommend that |
Sci J, 1995, China | centre, over 20 years. | pain, dyspnoea, cough, | asymptomatic | |
[15] | Included paediatric | fever, infection, | bronchogenic cysts | |
patients, but not | dysphagia, haemoptysis) | should be excised | ||
Retrospective | specified how many | because of the high risk | ||
cohort study (2b) | Preoperative | 36.4% diagnosed with | of complications, | |
Mean postoperative | diagnosis | CT | although this conclusion | |
follow-up seven years | was not tested in the | |||
Postoperative | No recurrence. | study | ||
complications | No complications. | |||
No mortality | ||||
Takeda et al., | 105 patients (with any | Presenting complaint | 40% symptomatic (chest | Heterogenous population. |
Chest, 2003, USA | mediastinal cyst) from | pain, dyspnoea, cough, | Data extracted for | |
[16] | one centre including | fever, sputum, | bronchogenic cysts only. | |
45 adults and six | dysphagia, haemoptysis) | Three patients had | ||
Retropective cohort | paediatric patients with | complicated surgery due | ||
study (2b) | bronchogenic cysts, | Preoperative | Could not extract data | to peri-cystic adhesions. |
over 50-year period | diagnosis | but improved diagnostic | Preventative resection | |
capabilities of MRI | preferred because of | |||
acknowledged | unpredictable clinical | |||
behaviour | ||||
Postoperative | Could not extract data | |||
complications | but described as | |||
‘acceptable’ by authors | ||||
Complications of | Two patients refused | |||
conservative | treatment: outcome not | |||
management | described | |||
Gursoy et al., Saudi | 28 adult patients with | Presenting complaint | 71% symptomatic | Six additional patients |
Med J, 2009, | preoperative diagnosis | (dyspnoea, chest pain, | excluded as preoperative | |
Turkey, [17] | of bronchogenic cyst | cough, fever) | diagnosis of | |
from one centre over | bronchogenic cyst not | |||
Retrospective | seven years | Preoperative | 82.4% correctly | confirmed histologically |
cohort study (2b) | diagnosis | diagnosed with CT | ||
36-month mean | Surgical resection | |||
postoperative | Postoperative | 11% early complication | recommended in | |
follow-up | complications | rate (wound infection, | asymptomatic patients | |
prolonged air leak, | because of the possibility | |||
pneumoperitoneum). | of malignant | |||
7% late complication | transformation and | |||
(dyspnea, | anatomic complications | |||
pneumothorax). | of delayed surgery | |||
No mortality | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Liu et al., Chin Med | 50 adult patients with | Presenting complaint | 66% symptomatic | Recommend surgical |
Sci J, 2009, China, | histopathologically | (cough, chest pain, | resection to confirm | |
[18] | proven bronchogenic | hemoptysis, dyspnoea, | diagnosis, avoid | |
cyst from one centre of | fever, dysphagia, | development of | ||
Retrospective | 24 years | paralysis, hoarseness). | symptoms or malignant | |
cohort study (2b) | 26% serious | change. Conclusions | ||
6.5-year mean | complications | drawn upon findings of | ||
follow-up | other studies | |||
Preoperative | 40% diagnosed | |||
diagnosis | preoperatively. | |||
14% misdiagnosed after | ||||
all investigations | ||||
Postoperative | 4% early complication | |||
complications | (persistent air leak, | |||
hoarseness). | ||||
No late complications. | ||||
No mortality | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Kosar et al., Heart | 29 patients from one | Presenting complaint | 86% symptomatic | The authors suggest |
Lung Circ, 2009 | centre, including 13 | (cough, sputum, pain, | there is a ‘general | |
Turkey, [19] | paediatric patients, | breathlessness, | consensus’ that all | |
over 15 years treated | haemoptysis, fever) | bronchogenic cysts | ||
Retrospective | with either resection | should be operated on to | ||
cohort study (2b) | or de-epithelialisation | Preoperative | 89.7% diagnosed | avoid development of |
diagnosis | preoperatively using | symptoms or | ||
predominantly CT | complications | |||
Postoperative | 17% early complication | |||
complications | rate (pneumonia, wound | |||
infections, prolonged air | ||||
leak); higher in | ||||
complicated cysts. | ||||
No late complications. | ||||
No mortality. | ||||
Recurrence in | ||||
de-epithelialised group | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Limaïauiem et al., | 33 patients from one | Presenting complaint | 94% symptomatic (chest | Management of all |
Lung, 2008, | centre over six years | pain, cough, | bronchogenic cysts | |
Tunisia, [20] | haemoptysis, dyspnoea, | based on complete | ||
Follow-up between | fever, dysphagia) | surgical excision. | ||
Retrospective | one and 51 months | Conclude that definitive | ||
cohort study (2b) | Preoperative | Correct diagnosis in | diagnosis is by histology | |
diagnosis | 33.3% | only and that complete | ||
surgical excision is | ||||
Postoperative | 14% Early complications | mandatory, although | ||
complications | (pneumothorax, | conclusions based on | ||
haemorrhage, pleural | findings of other studies | |||
effusion, seizure). | ||||
No late complications. | ||||
No mortality | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Granato et al., | 30 adult patients treated | Presenting complaint | 30% symptomatic | Two symptomatic cysts |
Asian Cardiovasc | for bronchogenic cysts | (cough, sputum, pain, | complicated | |
Thorac Ann, 2009, | from one centre, over | fever, weakness) | intraoperatively by severe | |
Italy, [21] | 32 years | adhesions | ||
Preoperative | Correct diagnosis in | |||
Retrospective | diagnosis | 100% (CT or CT and | One case of large-cell | |
cohort study (2b) | MRI) | anaplastic carcinoma in | ||
wall of cyst | ||||
Postoperative | 10% intraoperative | |||
complications | complications. | Excision of | ||
10% postoperative | asymptomatic cysts | |||
complications | advocated to avoid | |||
complex surgery and | ||||
Complications of | No patients | complications, and also | ||
conservative | conservatively managed | to reduce malignant | ||
management | potential | |||
De Giacomo et al., | 30 adult patients from | Presenting complaint | 37% symptomatic | Authors feel that patients |
Eur J Cardiothorac Surg | one centre over | (cough, pain, dysphagia) | cannot be completely | |
2009, Italy, [22] | 12 years | assured about | ||
Postoperative | None | conservative | ||
Retrospective | Follow-up | complications | management but | |
cohort study (2b) | 3–120 months | acknowledge the | ||
Complications of | Asymptomatic patients | management is | ||
conservative | requested surgery | controversial | ||
management | because of enlarging | |||
cysts, risk of | ||||
complication or fear of | ||||
malignancy | ||||
Costa Júnior Ada | 60 patients with | Presenting complaint | 92% symptomatic | Heterogenous group of |
et al., J Bras | pulmonary | (cough, dyspnoea, pain, | patients and disorders. | |
Pneumol, 2008, | malformations | infection) | Difficult to extract | |
Brazil, [23] | (including 27 with | bronchogenic cyst data | ||
bronchogenic cyst) | Preoperative | ‘Frequent’ misdiagnosis | ||
Retrospective | from one centre | diagnosis | Prognosis noted to be | |
cohort study (2b) | over 35 years, | unpredictable. One | ||
including 40 paediatric | Postoperative | 23% (pneumonia, | patient found to have | |
patients | complications | atelectasis, empyema, | adenocarcinoma in wall | |
sepsis). | of cyst | |||
3.3% mortality | ||||
Complications of | Diagnosis/treatment | |||
conservative | delayed in three patients | |||
management | up to 36 months (mean | |||
15): outcome in these | ||||
patients not specified | ||||
Weber et al., | 12 patients from a | Presenting complaint | 42% symptomatic | Agree that management |
Ann Thorac Surg, | single centre | (cough, pain, pneumonia) | of asymptomatic cysts is | |
2004, Switzerland, | undergoing | controversial but that | ||
[24] | video-assisted | Preoperative | 100% correct diagnosis | there appears to be no |
thoracoscopic surgery | diagnosis | with CT with or without | need for urgent surgery | |
Retrospective | for bronchogenic | MRI. MRI noted to be | in these cases provided | |
cohort study (2b) | cysts, over seven years | superior | that a simple cyst has | |
been clearly diagnosed | ||||
40.5-month mean | Postoperative | None | ||
follow-up | complications | |||
Complications of | Six patients observed for | |||
conservative | between two and | |||
management | 22 years without | |||
complications.Three | ||||
developed mild | ||||
symptoms. | ||||
Three patients eventually | ||||
requested surgery | ||||
because of fear of | ||||
malignancy/complications | ||||
or enlarging cyst |
CT, computed tomography; EUS, endoscopic ultrasound; USS, ultrasound scan; FNA, fine needle aspiration.
Author, date and country | Patient group | Outcomes | Key results | Comments |
Study type | ||||
(level of evidence) | ||||
Laberge et al., | Review of literature | Presenting complaint | Dysphagia, dyspnoea, | Highly heterogenous |
Semin Pediatr Surg, | regarding asymptomatic | infection, haemoptysis | group including all adult | |
2005, Canada, [2] | congenital lung | and haemothorax (no | and paediatric congenital | |
malformations, | figures of incidence) | lung malformations | ||
Review (2a) | including bronchogenic | |||
cysts, in a paediatric | Postoperative | Adult postoperative | Two cases of | |
population | complications | complications not | malignancy in | |
discussed | bronchogenic cysts | |||
Includes papers on adult | noted in adult patients | |||
congenital lung | Complications of | One adult patient with | ||
malformations | conservative | abnormal chest X-ray | Conclusions based on | |
management | eventually presented | management of | ||
10 years later with | paediatric malformations | |||
dyspnoea and was found | but recommend early | |||
to have | excision of bronchogenic | |||
bronchioloalveolar | cysts due to progression | |||
carcinoma associated | to symptoms, malignant | |||
with bronchogenic cyst. | potential and | |||
Another patient had ‘long | complications of | |||
standing’ history of cyst | symptomatic cysts | |||
infections and later found | ||||
to have associated | ||||
mesenchymal malignancy | ||||
Sarper et al., Tex | 22 patients from one | Presenting complaint | 45% presented with | Recommended surgical |
Heart Inst J, 2003, | centre, over 15 years | severe haemoptysis, | resection of all suspected | |
Turkey, [3] | pneumothorax and | bronchogenic cysts in | ||
5.2-year median | pleuritis, oesophageal | operable candidates due | ||
Retrospective | postoperative follow-up | compression, infected | to difficulties in | |
cohort study (2b) | cyst, or postobstructive | establishing definitive | ||
pneumonia. | diagnoses and frequency | |||
82% presented with | of complications | |||
symptoms of cough, | ||||
pain, dyspnoea, | ||||
dysphagia or infection | ||||
Postoperative | 5% (one patient) | |||
complications | persistent air leak | |||
No recurrence | ||||
No late sequelae | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Kanemitsu et al., | 17 patients (16 adults | Presenting complaint | 29% symptomatic (9% | It is noted that the |
Surg Today, 1999, | and one paediatric) from | of mediastinal cysts, 67% | advanced age of some | |
Japan, [4] | one centre, over | of intrapulmonary cysts) | patients at presentation | |
30 years | with cough, sputum, | suggests that these cysts | ||
Retrospective | fever, pain or weight loss | can remain | ||
cohort study (2b) | 30-month median | asymptomatic forever. | ||
postoperative follow-up | Preoperative | 69% diagnosed with CT | The authors state that the | |
diagnosis | 100% diagnosed with | most appropriate | ||
MRI | treatment for | |||
asymptomatic cysts is | ||||
Operative findings | 41% adhesions, 6% | controversial but due to | ||
complicated | diagnostic limitations and | |||
the possibility of | ||||
Postoperative | No postoperative | symptoms arising or | ||
complications | complications. | malignant transformation | ||
No recurrence | that surgery is advocated | |||
Complications of | None in one patient | |||
incomplete resection | where residual tissue | |||
ablated with | ||||
electro-cautery | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Cioffi et al., Chest, | 27 adults of whom | Presenting complaint | 50% chest pain, 13% | Conclude that all patients |
1998, Italy, [5] | 16 with bronchogenic | cough, 6% epigastric | should have surgical | |
cyst from one centre, | pain. | resection for definitive | ||
Retrospective | over 20 years | 44% asymptomatic | diagnosis and to | |
cohort study (2b) | (incidental finding) | minimise complications | ||
Four-year median | that might arise from | |||
postoperative follow-up | Preoperative | 100% preoperatively | symptomatic cysts | |
diagnosis | diagnosed with CT, EUS | |||
Postoperative | No postoperative | |||
complications | complications | |||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Aktogu et al., Eur | 31 patients (30 adults | Presenting complaint | 19% superior vena cava | Thirteen patients |
Respir J, 1996, | and one paediatric) from | syndrome, tracheal | symptomatic at the time | |
Turkey, [6] | one centre, over | compression, | of surgery had complex | |
19 years | pneumothorax, pleurisy | peri-cystic adhesions or | ||
Retrospective | or pneumonia. | fistulisation. | ||
cohort study (2b) | Follow-up 2–10 years | 81% cough, infection, | Surgical treatment of | |
postoperatively | pain, dyspnoea, anorexia/ | asymptomatic cysts is | ||
weight loss, haemoptysis | recommended to avoid | |||
potentially | ||||
Preoperative | 19% asymptomatic | life-threatening | ||
diagnosis | (incidental finding). | complications and for | ||
71% undiagnosed with | definitive diagnosis | |||
CT | ||||
Postoperative | No recurrence | |||
complications | ||||
Complications of | Two of six initially | |||
conservative | asymptomatic patients | |||
management | either had cyst | |||
enlargement or became | ||||
symptomatic | ||||
Ribet et al., Ann | 41 patients from one | Presenting complaint | 80% symptomatic | Uncertain what |
Thorac Surg, 1996, | centre, including 20 | (cough, pain, purulent | proportion of | |
France, [7] | paediatric cases, over | sputum, haemoptysis and | bronchogenic cysts | |
25 years | dyspnoea) in adult life | remain asymptomatic | ||
Retrospective | and long-term prognosis | |||
cohort study (2b) | 2.6-year mean | Preoperative | 45% undiagnosed | unpredictable. |
postoperative follow-up | diagnosis | Preventative surgery | ||
recommended | ||||
Postoperative | 5% (one patient) | |||
complications | bronchial fistula. | |||
No recurrence in 88% | ||||
(remainder lost to | ||||
follow-up). | ||||
No deaths | ||||
Complications of | 15% (three patients) | |||
conservative | followed for 11 months, | |||
management | five years and unknown | |||
length of time since | ||||
diagnosis developed no | ||||
symptoms but were | ||||
operated on. | ||||
15% (three patients) | ||||
initially with cough, | ||||
recurrent bronchitis and | ||||
no symptoms | ||||
(respectively) refused | ||||
surgery but lost to | ||||
follow-up | ||||
Cuypers et al., Eur | 20 adult patients from | Presenting complaint | 30% symptomatic | The authors found the |
J Cardiothorac | one centre, over | (pneumonia, abscess, | risk of malignancy and | |
Surg, 1996, | 18 years | dysphagia and cough) | cyst-related | |
Belgium, [8] | 70% asymptomatic | complications to be | ||
No long-term follow-up | justification for operative | |||
Retrospective | Preoperative | 25% undiagnosed after | treatment in all cases of | |
cohort study (2b) | diagnosis | CT, bronchoscopy, | bronchogenic cysts | |
barium swallow or echo | ||||
(all cases had CT with | ||||
dense fluid) | ||||
Postoperative | No postoperative | |||
complications | morbidity; one case | |||
histologically associated | ||||
with squamous cell | ||||
carcinoma | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Ribet et al., J | 69 patients from one | Presenting complaint | 63.7% symptomatic | Due to the frequency of |
Thorac Cardiovasc | centre, including 24 | pain, respiratory tract | late complications with | |
Surg, 1995, France, | paediatric cases, over | infection, cough, | asymptomatic cysts and | |
[9] | 25 years | dyspnoea, dysphagia, | the unpredictable | |
heartburn) | prognosis, preventative | |||
Retrospective | 4.2-year mean | surgery was | ||
cohort study (2b) | postoperative follow-up | Preoperative | 22% initially | recommended |
diagnosis | misdiagnosed (11% not | |||
as bronchogenic cyst; | ||||
11% as bronchogenic | ||||
cyst when actually cystic | ||||
neurogenic tumour, | ||||
benign lymphoma and | ||||
haemolymphangioma) | ||||
Postoperative | 13.4% postoperative | |||
complications | morbidity (infection, | |||
chylothorax and phrenic | ||||
paresis). | ||||
12% symptoms of pain | ||||
or dyspnoea | ||||
postoperative (7% | ||||
symptomatic | ||||
preoperative, 5% | ||||
asymptomatic) | ||||
Complications of | 11% refused operation, | |||
conservative | only 4% followed up | |||
management | (two patients): one | |||
remained asymptomatic, | ||||
one died of generalised | ||||
malignancy of unknown | ||||
origin. | ||||
7% had incomplete | ||||
operations but cyst | ||||
remained stable or had no | ||||
recurrence | ||||
Patel et al., Chest, | 18 adult patients from | Presenting complaint | 44% symptomatic | There was no statistical |
1994, USA, [10] | one centre, over | (cough, pain) of which | difference in the | |
19-year period | 11% serious (dyspnea, | frequency of | ||
Retrospective | infection). | intraoperative difficulties | ||
cohort study (2b) | Follow-up 12 | 56% asymptomatic | or postoperative | |
months to 11 years | complications between | |||
(mean not given) | Preoperative | 37.5% undiagnosed with | asymptomatic and | |
diagnosis | CT, angio, USS, FNA, | symptomatic patients. | ||
barium swallow, | The authors concluded, | |||
bronchoscopy or | however, that surgery is | |||
mediastinoscopy | advocated in | |||
Operative difficulties | No difference in | asymptomatic cysts due | ||
frequency of operative | to the potential for | |||
difficulties between | complications, incorrect | |||
symptomatic and | diagnosis or progression | |||
asymptomatic patients | to symptoms | |||
(P=0.0656) | ||||
Postoperative | No difference (P=0.596) | |||
complications | in complication rate | |||
between preoperatively | ||||
asymptomatic (14%) or | ||||
symptomatic (27%) | ||||
patients. Complications | ||||
included phrenic nerve | ||||
paresis. | ||||
11% delayed | ||||
complications | ||||
(oesophageal stricture | ||||
and recurrence) | ||||
Complications of | Three out of seven | |||
conservative | asymptomatic patients | |||
management | followed up developed | |||
symptoms; four patients | ||||
lost to follow-up | ||||
Suen et al., Ann | 42 patients from one | Presenting complaint | 50% symptomatic (pain, | Complete excision |
Thorac Surg, | centre, over 30 years | cough, fever, dysphagia, | recommended in most | |
1993, USA, [11] | purulent sputum, | cases to relieve | ||
haemoptysis, dyspnoea). | symptoms, prevent | |||
Retrospective | 26% complicated | complications and | ||
cohort study (2b) | (dysphagia, haemorrhage, | confirm diagnosis | ||
infection and one patient | ||||
with adenocarcinoma in | ||||
cyst) | ||||
Preoperative | 59% no preoperative | |||
diagnosis | diagnosis (more recent | |||
cases, better success | ||||
rate) | ||||
Postoperative | 5% postoperative | |||
complications | complications (wound | |||
infection and C. Diff | ||||
colitis). | ||||
No recurrence. | ||||
No deaths | ||||
Complications of | Two patients treated | |||
conservative | conservatively (one | |||
management | follow-up only and one | |||
drainage) with no | ||||
complications | ||||
St-Georges et al., | 86 patients from one | Presenting complaint | 72% patients | 35/86 (41%) of patients |
Ann Thorac Surg, | centre, over 20 years | symptomatic by time of | operated on had major | |
1991, Canada, [12] | surgery (57% | operative difficulties: all | ||
progressive symptoms | these were patients with | |||
Retrospective | and 15% acute). | symptoms | ||
cohort study (2b) | 53% patients with >1 | preoperatively | ||
symptom (chest pain, | ||||
cough, dyspnoea, fever, | Resection recommended | |||
sputum, anorexia/weight | as majority of patients | |||
loss, dysphagia, | eventually develop | |||
haemoptysis). | symptoms or | |||
38% with complications | complications | |||
(fistula with airway, | ||||
ulceration cyst wall, | ||||
haemorrhage, infection, | ||||
bronchial atresia) | ||||
Preoperative | 57% patients presumed | |||
diagnosis | diagnosis at surgery after | |||
CT and angiography. | ||||
Positive diagnosis never | ||||
made preoperatively | ||||
Operative findings | 41% had complicated | |||
cysts (fistula, ulcer, | ||||
haemorrhage, infection, | ||||
atresia) at operation | ||||
Postoperative | 11% intraoperative | |||
complications | complications (vagal | |||
trunk division, segmental | ||||
bronchus laceration, | ||||
oesophageal mucosal | ||||
laceration). | ||||
9% postoperative | ||||
complications | ||||
(atelectasis, pleural | ||||
effusion, wound | ||||
infection, transient | ||||
Horner's syndrome, | ||||
respiratory failure | ||||
requiring tracheostomy | ||||
and haemothorax) with | ||||
major postoperative | ||||
complications in | ||||
symptomatic patients | ||||
Complications of | 37 patients were | |||
conservative | followed up | |||
management | conservatively, and | |||
13 (35%) were still | ||||
asymptomatic at time of | ||||
operation | ||||
Cartmill and | 20 patients from one | Presenting complaint | 75% symptomatic (chest | Advocate surgery for |
Hughes, Aust N Z J | centre, over 10 years. | pain, cough, | symptom relief, | |
Surg, 1989, | Included some | haemoptysis, dysphagia). | exclusion of malignancy | |
Australia, [13] | paediatric patients (not | 5% serious complications | and prevention of | |
known how many) | catastrophic | |||
Retrospective | Preoperative | 45% diagnosed with CT, | complications | |
cohort study (2b) | Mean postoperative | diagnosis | barium swallow and | |
follow-up 67 months | aortography | |||
Postoperative | 5% (one patient) multiple | |||
complications | PE. | |||
No recurrence. | ||||
No death | ||||
Complications of | 5% (one patient) declined | |||
conservative | surgery initially but | |||
management | returned for elective | |||
treatment after six years. | ||||
Reason not given | ||||
Coselli et al., Ann | Eight patients from one | Presenting complaint | 75% symptomatic | Recommend excision to |
Thorac Surg, 1987, | centre, over 11 years | (dysphagia, epigastric | establish diagnosis, | |
USA, [14] | pain, respiratory distress, | alleviate symptoms and | ||
dyspnoea, chest pain) | prevent complications | |||
Case series (4) | ||||
Preoperative | With use of CT; operated | |||
diagnosis | on to confirm diagnosis | |||
Operative findings | Previously infected cysts | |||
more difficult and | ||||
hazardous to excise | ||||
Ge et al., Chin Med | 22 patients from one | Presenting complaint | 91% symptomatic (chest | Recommend that |
Sci J, 1995, China | centre, over 20 years. | pain, dyspnoea, cough, | asymptomatic | |
[15] | Included paediatric | fever, infection, | bronchogenic cysts | |
patients, but not | dysphagia, haemoptysis) | should be excised | ||
Retrospective | specified how many | because of the high risk | ||
cohort study (2b) | Preoperative | 36.4% diagnosed with | of complications, | |
Mean postoperative | diagnosis | CT | although this conclusion | |
follow-up seven years | was not tested in the | |||
Postoperative | No recurrence. | study | ||
complications | No complications. | |||
No mortality | ||||
Takeda et al., | 105 patients (with any | Presenting complaint | 40% symptomatic (chest | Heterogenous population. |
Chest, 2003, USA | mediastinal cyst) from | pain, dyspnoea, cough, | Data extracted for | |
[16] | one centre including | fever, sputum, | bronchogenic cysts only. | |
45 adults and six | dysphagia, haemoptysis) | Three patients had | ||
Retropective cohort | paediatric patients with | complicated surgery due | ||
study (2b) | bronchogenic cysts, | Preoperative | Could not extract data | to peri-cystic adhesions. |
over 50-year period | diagnosis | but improved diagnostic | Preventative resection | |
capabilities of MRI | preferred because of | |||
acknowledged | unpredictable clinical | |||
behaviour | ||||
Postoperative | Could not extract data | |||
complications | but described as | |||
‘acceptable’ by authors | ||||
Complications of | Two patients refused | |||
conservative | treatment: outcome not | |||
management | described | |||
Gursoy et al., Saudi | 28 adult patients with | Presenting complaint | 71% symptomatic | Six additional patients |
Med J, 2009, | preoperative diagnosis | (dyspnoea, chest pain, | excluded as preoperative | |
Turkey, [17] | of bronchogenic cyst | cough, fever) | diagnosis of | |
from one centre over | bronchogenic cyst not | |||
Retrospective | seven years | Preoperative | 82.4% correctly | confirmed histologically |
cohort study (2b) | diagnosis | diagnosed with CT | ||
36-month mean | Surgical resection | |||
postoperative | Postoperative | 11% early complication | recommended in | |
follow-up | complications | rate (wound infection, | asymptomatic patients | |
prolonged air leak, | because of the possibility | |||
pneumoperitoneum). | of malignant | |||
7% late complication | transformation and | |||
(dyspnea, | anatomic complications | |||
pneumothorax). | of delayed surgery | |||
No mortality | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Liu et al., Chin Med | 50 adult patients with | Presenting complaint | 66% symptomatic | Recommend surgical |
Sci J, 2009, China, | histopathologically | (cough, chest pain, | resection to confirm | |
[18] | proven bronchogenic | hemoptysis, dyspnoea, | diagnosis, avoid | |
cyst from one centre of | fever, dysphagia, | development of | ||
Retrospective | 24 years | paralysis, hoarseness). | symptoms or malignant | |
cohort study (2b) | 26% serious | change. Conclusions | ||
6.5-year mean | complications | drawn upon findings of | ||
follow-up | other studies | |||
Preoperative | 40% diagnosed | |||
diagnosis | preoperatively. | |||
14% misdiagnosed after | ||||
all investigations | ||||
Postoperative | 4% early complication | |||
complications | (persistent air leak, | |||
hoarseness). | ||||
No late complications. | ||||
No mortality | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Kosar et al., Heart | 29 patients from one | Presenting complaint | 86% symptomatic | The authors suggest |
Lung Circ, 2009 | centre, including 13 | (cough, sputum, pain, | there is a ‘general | |
Turkey, [19] | paediatric patients, | breathlessness, | consensus’ that all | |
over 15 years treated | haemoptysis, fever) | bronchogenic cysts | ||
Retrospective | with either resection | should be operated on to | ||
cohort study (2b) | or de-epithelialisation | Preoperative | 89.7% diagnosed | avoid development of |
diagnosis | preoperatively using | symptoms or | ||
predominantly CT | complications | |||
Postoperative | 17% early complication | |||
complications | rate (pneumonia, wound | |||
infections, prolonged air | ||||
leak); higher in | ||||
complicated cysts. | ||||
No late complications. | ||||
No mortality. | ||||
Recurrence in | ||||
de-epithelialised group | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Limaïauiem et al., | 33 patients from one | Presenting complaint | 94% symptomatic (chest | Management of all |
Lung, 2008, | centre over six years | pain, cough, | bronchogenic cysts | |
Tunisia, [20] | haemoptysis, dyspnoea, | based on complete | ||
Follow-up between | fever, dysphagia) | surgical excision. | ||
Retrospective | one and 51 months | Conclude that definitive | ||
cohort study (2b) | Preoperative | Correct diagnosis in | diagnosis is by histology | |
diagnosis | 33.3% | only and that complete | ||
surgical excision is | ||||
Postoperative | 14% Early complications | mandatory, although | ||
complications | (pneumothorax, | conclusions based on | ||
haemorrhage, pleural | findings of other studies | |||
effusion, seizure). | ||||
No late complications. | ||||
No mortality | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Granato et al., | 30 adult patients treated | Presenting complaint | 30% symptomatic | Two symptomatic cysts |
Asian Cardiovasc | for bronchogenic cysts | (cough, sputum, pain, | complicated | |
Thorac Ann, 2009, | from one centre, over | fever, weakness) | intraoperatively by severe | |
Italy, [21] | 32 years | adhesions | ||
Preoperative | Correct diagnosis in | |||
Retrospective | diagnosis | 100% (CT or CT and | One case of large-cell | |
cohort study (2b) | MRI) | anaplastic carcinoma in | ||
wall of cyst | ||||
Postoperative | 10% intraoperative | |||
complications | complications. | Excision of | ||
10% postoperative | asymptomatic cysts | |||
complications | advocated to avoid | |||
complex surgery and | ||||
Complications of | No patients | complications, and also | ||
conservative | conservatively managed | to reduce malignant | ||
management | potential | |||
De Giacomo et al., | 30 adult patients from | Presenting complaint | 37% symptomatic | Authors feel that patients |
Eur J Cardiothorac Surg | one centre over | (cough, pain, dysphagia) | cannot be completely | |
2009, Italy, [22] | 12 years | assured about | ||
Postoperative | None | conservative | ||
Retrospective | Follow-up | complications | management but | |
cohort study (2b) | 3–120 months | acknowledge the | ||
Complications of | Asymptomatic patients | management is | ||
conservative | requested surgery | controversial | ||
management | because of enlarging | |||
cysts, risk of | ||||
complication or fear of | ||||
malignancy | ||||
Costa Júnior Ada | 60 patients with | Presenting complaint | 92% symptomatic | Heterogenous group of |
et al., J Bras | pulmonary | (cough, dyspnoea, pain, | patients and disorders. | |
Pneumol, 2008, | malformations | infection) | Difficult to extract | |
Brazil, [23] | (including 27 with | bronchogenic cyst data | ||
bronchogenic cyst) | Preoperative | ‘Frequent’ misdiagnosis | ||
Retrospective | from one centre | diagnosis | Prognosis noted to be | |
cohort study (2b) | over 35 years, | unpredictable. One | ||
including 40 paediatric | Postoperative | 23% (pneumonia, | patient found to have | |
patients | complications | atelectasis, empyema, | adenocarcinoma in wall | |
sepsis). | of cyst | |||
3.3% mortality | ||||
Complications of | Diagnosis/treatment | |||
conservative | delayed in three patients | |||
management | up to 36 months (mean | |||
15): outcome in these | ||||
patients not specified | ||||
Weber et al., | 12 patients from a | Presenting complaint | 42% symptomatic | Agree that management |
Ann Thorac Surg, | single centre | (cough, pain, pneumonia) | of asymptomatic cysts is | |
2004, Switzerland, | undergoing | controversial but that | ||
[24] | video-assisted | Preoperative | 100% correct diagnosis | there appears to be no |
thoracoscopic surgery | diagnosis | with CT with or without | need for urgent surgery | |
Retrospective | for bronchogenic | MRI. MRI noted to be | in these cases provided | |
cohort study (2b) | cysts, over seven years | superior | that a simple cyst has | |
been clearly diagnosed | ||||
40.5-month mean | Postoperative | None | ||
follow-up | complications | |||
Complications of | Six patients observed for | |||
conservative | between two and | |||
management | 22 years without | |||
complications.Three | ||||
developed mild | ||||
symptoms. | ||||
Three patients eventually | ||||
requested surgery | ||||
because of fear of | ||||
malignancy/complications | ||||
or enlarging cyst |
Author, date and country | Patient group | Outcomes | Key results | Comments |
Study type | ||||
(level of evidence) | ||||
Laberge et al., | Review of literature | Presenting complaint | Dysphagia, dyspnoea, | Highly heterogenous |
Semin Pediatr Surg, | regarding asymptomatic | infection, haemoptysis | group including all adult | |
2005, Canada, [2] | congenital lung | and haemothorax (no | and paediatric congenital | |
malformations, | figures of incidence) | lung malformations | ||
Review (2a) | including bronchogenic | |||
cysts, in a paediatric | Postoperative | Adult postoperative | Two cases of | |
population | complications | complications not | malignancy in | |
discussed | bronchogenic cysts | |||
Includes papers on adult | noted in adult patients | |||
congenital lung | Complications of | One adult patient with | ||
malformations | conservative | abnormal chest X-ray | Conclusions based on | |
management | eventually presented | management of | ||
10 years later with | paediatric malformations | |||
dyspnoea and was found | but recommend early | |||
to have | excision of bronchogenic | |||
bronchioloalveolar | cysts due to progression | |||
carcinoma associated | to symptoms, malignant | |||
with bronchogenic cyst. | potential and | |||
Another patient had ‘long | complications of | |||
standing’ history of cyst | symptomatic cysts | |||
infections and later found | ||||
to have associated | ||||
mesenchymal malignancy | ||||
Sarper et al., Tex | 22 patients from one | Presenting complaint | 45% presented with | Recommended surgical |
Heart Inst J, 2003, | centre, over 15 years | severe haemoptysis, | resection of all suspected | |
Turkey, [3] | pneumothorax and | bronchogenic cysts in | ||
5.2-year median | pleuritis, oesophageal | operable candidates due | ||
Retrospective | postoperative follow-up | compression, infected | to difficulties in | |
cohort study (2b) | cyst, or postobstructive | establishing definitive | ||
pneumonia. | diagnoses and frequency | |||
82% presented with | of complications | |||
symptoms of cough, | ||||
pain, dyspnoea, | ||||
dysphagia or infection | ||||
Postoperative | 5% (one patient) | |||
complications | persistent air leak | |||
No recurrence | ||||
No late sequelae | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Kanemitsu et al., | 17 patients (16 adults | Presenting complaint | 29% symptomatic (9% | It is noted that the |
Surg Today, 1999, | and one paediatric) from | of mediastinal cysts, 67% | advanced age of some | |
Japan, [4] | one centre, over | of intrapulmonary cysts) | patients at presentation | |
30 years | with cough, sputum, | suggests that these cysts | ||
Retrospective | fever, pain or weight loss | can remain | ||
cohort study (2b) | 30-month median | asymptomatic forever. | ||
postoperative follow-up | Preoperative | 69% diagnosed with CT | The authors state that the | |
diagnosis | 100% diagnosed with | most appropriate | ||
MRI | treatment for | |||
asymptomatic cysts is | ||||
Operative findings | 41% adhesions, 6% | controversial but due to | ||
complicated | diagnostic limitations and | |||
the possibility of | ||||
Postoperative | No postoperative | symptoms arising or | ||
complications | complications. | malignant transformation | ||
No recurrence | that surgery is advocated | |||
Complications of | None in one patient | |||
incomplete resection | where residual tissue | |||
ablated with | ||||
electro-cautery | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Cioffi et al., Chest, | 27 adults of whom | Presenting complaint | 50% chest pain, 13% | Conclude that all patients |
1998, Italy, [5] | 16 with bronchogenic | cough, 6% epigastric | should have surgical | |
cyst from one centre, | pain. | resection for definitive | ||
Retrospective | over 20 years | 44% asymptomatic | diagnosis and to | |
cohort study (2b) | (incidental finding) | minimise complications | ||
Four-year median | that might arise from | |||
postoperative follow-up | Preoperative | 100% preoperatively | symptomatic cysts | |
diagnosis | diagnosed with CT, EUS | |||
Postoperative | No postoperative | |||
complications | complications | |||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Aktogu et al., Eur | 31 patients (30 adults | Presenting complaint | 19% superior vena cava | Thirteen patients |
Respir J, 1996, | and one paediatric) from | syndrome, tracheal | symptomatic at the time | |
Turkey, [6] | one centre, over | compression, | of surgery had complex | |
19 years | pneumothorax, pleurisy | peri-cystic adhesions or | ||
Retrospective | or pneumonia. | fistulisation. | ||
cohort study (2b) | Follow-up 2–10 years | 81% cough, infection, | Surgical treatment of | |
postoperatively | pain, dyspnoea, anorexia/ | asymptomatic cysts is | ||
weight loss, haemoptysis | recommended to avoid | |||
potentially | ||||
Preoperative | 19% asymptomatic | life-threatening | ||
diagnosis | (incidental finding). | complications and for | ||
71% undiagnosed with | definitive diagnosis | |||
CT | ||||
Postoperative | No recurrence | |||
complications | ||||
Complications of | Two of six initially | |||
conservative | asymptomatic patients | |||
management | either had cyst | |||
enlargement or became | ||||
symptomatic | ||||
Ribet et al., Ann | 41 patients from one | Presenting complaint | 80% symptomatic | Uncertain what |
Thorac Surg, 1996, | centre, including 20 | (cough, pain, purulent | proportion of | |
France, [7] | paediatric cases, over | sputum, haemoptysis and | bronchogenic cysts | |
25 years | dyspnoea) in adult life | remain asymptomatic | ||
Retrospective | and long-term prognosis | |||
cohort study (2b) | 2.6-year mean | Preoperative | 45% undiagnosed | unpredictable. |
postoperative follow-up | diagnosis | Preventative surgery | ||
recommended | ||||
Postoperative | 5% (one patient) | |||
complications | bronchial fistula. | |||
No recurrence in 88% | ||||
(remainder lost to | ||||
follow-up). | ||||
No deaths | ||||
Complications of | 15% (three patients) | |||
conservative | followed for 11 months, | |||
management | five years and unknown | |||
length of time since | ||||
diagnosis developed no | ||||
symptoms but were | ||||
operated on. | ||||
15% (three patients) | ||||
initially with cough, | ||||
recurrent bronchitis and | ||||
no symptoms | ||||
(respectively) refused | ||||
surgery but lost to | ||||
follow-up | ||||
Cuypers et al., Eur | 20 adult patients from | Presenting complaint | 30% symptomatic | The authors found the |
J Cardiothorac | one centre, over | (pneumonia, abscess, | risk of malignancy and | |
Surg, 1996, | 18 years | dysphagia and cough) | cyst-related | |
Belgium, [8] | 70% asymptomatic | complications to be | ||
No long-term follow-up | justification for operative | |||
Retrospective | Preoperative | 25% undiagnosed after | treatment in all cases of | |
cohort study (2b) | diagnosis | CT, bronchoscopy, | bronchogenic cysts | |
barium swallow or echo | ||||
(all cases had CT with | ||||
dense fluid) | ||||
Postoperative | No postoperative | |||
complications | morbidity; one case | |||
histologically associated | ||||
with squamous cell | ||||
carcinoma | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Ribet et al., J | 69 patients from one | Presenting complaint | 63.7% symptomatic | Due to the frequency of |
Thorac Cardiovasc | centre, including 24 | pain, respiratory tract | late complications with | |
Surg, 1995, France, | paediatric cases, over | infection, cough, | asymptomatic cysts and | |
[9] | 25 years | dyspnoea, dysphagia, | the unpredictable | |
heartburn) | prognosis, preventative | |||
Retrospective | 4.2-year mean | surgery was | ||
cohort study (2b) | postoperative follow-up | Preoperative | 22% initially | recommended |
diagnosis | misdiagnosed (11% not | |||
as bronchogenic cyst; | ||||
11% as bronchogenic | ||||
cyst when actually cystic | ||||
neurogenic tumour, | ||||
benign lymphoma and | ||||
haemolymphangioma) | ||||
Postoperative | 13.4% postoperative | |||
complications | morbidity (infection, | |||
chylothorax and phrenic | ||||
paresis). | ||||
12% symptoms of pain | ||||
or dyspnoea | ||||
postoperative (7% | ||||
symptomatic | ||||
preoperative, 5% | ||||
asymptomatic) | ||||
Complications of | 11% refused operation, | |||
conservative | only 4% followed up | |||
management | (two patients): one | |||
remained asymptomatic, | ||||
one died of generalised | ||||
malignancy of unknown | ||||
origin. | ||||
7% had incomplete | ||||
operations but cyst | ||||
remained stable or had no | ||||
recurrence | ||||
Patel et al., Chest, | 18 adult patients from | Presenting complaint | 44% symptomatic | There was no statistical |
1994, USA, [10] | one centre, over | (cough, pain) of which | difference in the | |
19-year period | 11% serious (dyspnea, | frequency of | ||
Retrospective | infection). | intraoperative difficulties | ||
cohort study (2b) | Follow-up 12 | 56% asymptomatic | or postoperative | |
months to 11 years | complications between | |||
(mean not given) | Preoperative | 37.5% undiagnosed with | asymptomatic and | |
diagnosis | CT, angio, USS, FNA, | symptomatic patients. | ||
barium swallow, | The authors concluded, | |||
bronchoscopy or | however, that surgery is | |||
mediastinoscopy | advocated in | |||
Operative difficulties | No difference in | asymptomatic cysts due | ||
frequency of operative | to the potential for | |||
difficulties between | complications, incorrect | |||
symptomatic and | diagnosis or progression | |||
asymptomatic patients | to symptoms | |||
(P=0.0656) | ||||
Postoperative | No difference (P=0.596) | |||
complications | in complication rate | |||
between preoperatively | ||||
asymptomatic (14%) or | ||||
symptomatic (27%) | ||||
patients. Complications | ||||
included phrenic nerve | ||||
paresis. | ||||
11% delayed | ||||
complications | ||||
(oesophageal stricture | ||||
and recurrence) | ||||
Complications of | Three out of seven | |||
conservative | asymptomatic patients | |||
management | followed up developed | |||
symptoms; four patients | ||||
lost to follow-up | ||||
Suen et al., Ann | 42 patients from one | Presenting complaint | 50% symptomatic (pain, | Complete excision |
Thorac Surg, | centre, over 30 years | cough, fever, dysphagia, | recommended in most | |
1993, USA, [11] | purulent sputum, | cases to relieve | ||
haemoptysis, dyspnoea). | symptoms, prevent | |||
Retrospective | 26% complicated | complications and | ||
cohort study (2b) | (dysphagia, haemorrhage, | confirm diagnosis | ||
infection and one patient | ||||
with adenocarcinoma in | ||||
cyst) | ||||
Preoperative | 59% no preoperative | |||
diagnosis | diagnosis (more recent | |||
cases, better success | ||||
rate) | ||||
Postoperative | 5% postoperative | |||
complications | complications (wound | |||
infection and C. Diff | ||||
colitis). | ||||
No recurrence. | ||||
No deaths | ||||
Complications of | Two patients treated | |||
conservative | conservatively (one | |||
management | follow-up only and one | |||
drainage) with no | ||||
complications | ||||
St-Georges et al., | 86 patients from one | Presenting complaint | 72% patients | 35/86 (41%) of patients |
Ann Thorac Surg, | centre, over 20 years | symptomatic by time of | operated on had major | |
1991, Canada, [12] | surgery (57% | operative difficulties: all | ||
progressive symptoms | these were patients with | |||
Retrospective | and 15% acute). | symptoms | ||
cohort study (2b) | 53% patients with >1 | preoperatively | ||
symptom (chest pain, | ||||
cough, dyspnoea, fever, | Resection recommended | |||
sputum, anorexia/weight | as majority of patients | |||
loss, dysphagia, | eventually develop | |||
haemoptysis). | symptoms or | |||
38% with complications | complications | |||
(fistula with airway, | ||||
ulceration cyst wall, | ||||
haemorrhage, infection, | ||||
bronchial atresia) | ||||
Preoperative | 57% patients presumed | |||
diagnosis | diagnosis at surgery after | |||
CT and angiography. | ||||
Positive diagnosis never | ||||
made preoperatively | ||||
Operative findings | 41% had complicated | |||
cysts (fistula, ulcer, | ||||
haemorrhage, infection, | ||||
atresia) at operation | ||||
Postoperative | 11% intraoperative | |||
complications | complications (vagal | |||
trunk division, segmental | ||||
bronchus laceration, | ||||
oesophageal mucosal | ||||
laceration). | ||||
9% postoperative | ||||
complications | ||||
(atelectasis, pleural | ||||
effusion, wound | ||||
infection, transient | ||||
Horner's syndrome, | ||||
respiratory failure | ||||
requiring tracheostomy | ||||
and haemothorax) with | ||||
major postoperative | ||||
complications in | ||||
symptomatic patients | ||||
Complications of | 37 patients were | |||
conservative | followed up | |||
management | conservatively, and | |||
13 (35%) were still | ||||
asymptomatic at time of | ||||
operation | ||||
Cartmill and | 20 patients from one | Presenting complaint | 75% symptomatic (chest | Advocate surgery for |
Hughes, Aust N Z J | centre, over 10 years. | pain, cough, | symptom relief, | |
Surg, 1989, | Included some | haemoptysis, dysphagia). | exclusion of malignancy | |
Australia, [13] | paediatric patients (not | 5% serious complications | and prevention of | |
known how many) | catastrophic | |||
Retrospective | Preoperative | 45% diagnosed with CT, | complications | |
cohort study (2b) | Mean postoperative | diagnosis | barium swallow and | |
follow-up 67 months | aortography | |||
Postoperative | 5% (one patient) multiple | |||
complications | PE. | |||
No recurrence. | ||||
No death | ||||
Complications of | 5% (one patient) declined | |||
conservative | surgery initially but | |||
management | returned for elective | |||
treatment after six years. | ||||
Reason not given | ||||
Coselli et al., Ann | Eight patients from one | Presenting complaint | 75% symptomatic | Recommend excision to |
Thorac Surg, 1987, | centre, over 11 years | (dysphagia, epigastric | establish diagnosis, | |
USA, [14] | pain, respiratory distress, | alleviate symptoms and | ||
dyspnoea, chest pain) | prevent complications | |||
Case series (4) | ||||
Preoperative | With use of CT; operated | |||
diagnosis | on to confirm diagnosis | |||
Operative findings | Previously infected cysts | |||
more difficult and | ||||
hazardous to excise | ||||
Ge et al., Chin Med | 22 patients from one | Presenting complaint | 91% symptomatic (chest | Recommend that |
Sci J, 1995, China | centre, over 20 years. | pain, dyspnoea, cough, | asymptomatic | |
[15] | Included paediatric | fever, infection, | bronchogenic cysts | |
patients, but not | dysphagia, haemoptysis) | should be excised | ||
Retrospective | specified how many | because of the high risk | ||
cohort study (2b) | Preoperative | 36.4% diagnosed with | of complications, | |
Mean postoperative | diagnosis | CT | although this conclusion | |
follow-up seven years | was not tested in the | |||
Postoperative | No recurrence. | study | ||
complications | No complications. | |||
No mortality | ||||
Takeda et al., | 105 patients (with any | Presenting complaint | 40% symptomatic (chest | Heterogenous population. |
Chest, 2003, USA | mediastinal cyst) from | pain, dyspnoea, cough, | Data extracted for | |
[16] | one centre including | fever, sputum, | bronchogenic cysts only. | |
45 adults and six | dysphagia, haemoptysis) | Three patients had | ||
Retropective cohort | paediatric patients with | complicated surgery due | ||
study (2b) | bronchogenic cysts, | Preoperative | Could not extract data | to peri-cystic adhesions. |
over 50-year period | diagnosis | but improved diagnostic | Preventative resection | |
capabilities of MRI | preferred because of | |||
acknowledged | unpredictable clinical | |||
behaviour | ||||
Postoperative | Could not extract data | |||
complications | but described as | |||
‘acceptable’ by authors | ||||
Complications of | Two patients refused | |||
conservative | treatment: outcome not | |||
management | described | |||
Gursoy et al., Saudi | 28 adult patients with | Presenting complaint | 71% symptomatic | Six additional patients |
Med J, 2009, | preoperative diagnosis | (dyspnoea, chest pain, | excluded as preoperative | |
Turkey, [17] | of bronchogenic cyst | cough, fever) | diagnosis of | |
from one centre over | bronchogenic cyst not | |||
Retrospective | seven years | Preoperative | 82.4% correctly | confirmed histologically |
cohort study (2b) | diagnosis | diagnosed with CT | ||
36-month mean | Surgical resection | |||
postoperative | Postoperative | 11% early complication | recommended in | |
follow-up | complications | rate (wound infection, | asymptomatic patients | |
prolonged air leak, | because of the possibility | |||
pneumoperitoneum). | of malignant | |||
7% late complication | transformation and | |||
(dyspnea, | anatomic complications | |||
pneumothorax). | of delayed surgery | |||
No mortality | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Liu et al., Chin Med | 50 adult patients with | Presenting complaint | 66% symptomatic | Recommend surgical |
Sci J, 2009, China, | histopathologically | (cough, chest pain, | resection to confirm | |
[18] | proven bronchogenic | hemoptysis, dyspnoea, | diagnosis, avoid | |
cyst from one centre of | fever, dysphagia, | development of | ||
Retrospective | 24 years | paralysis, hoarseness). | symptoms or malignant | |
cohort study (2b) | 26% serious | change. Conclusions | ||
6.5-year mean | complications | drawn upon findings of | ||
follow-up | other studies | |||
Preoperative | 40% diagnosed | |||
diagnosis | preoperatively. | |||
14% misdiagnosed after | ||||
all investigations | ||||
Postoperative | 4% early complication | |||
complications | (persistent air leak, | |||
hoarseness). | ||||
No late complications. | ||||
No mortality | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Kosar et al., Heart | 29 patients from one | Presenting complaint | 86% symptomatic | The authors suggest |
Lung Circ, 2009 | centre, including 13 | (cough, sputum, pain, | there is a ‘general | |
Turkey, [19] | paediatric patients, | breathlessness, | consensus’ that all | |
over 15 years treated | haemoptysis, fever) | bronchogenic cysts | ||
Retrospective | with either resection | should be operated on to | ||
cohort study (2b) | or de-epithelialisation | Preoperative | 89.7% diagnosed | avoid development of |
diagnosis | preoperatively using | symptoms or | ||
predominantly CT | complications | |||
Postoperative | 17% early complication | |||
complications | rate (pneumonia, wound | |||
infections, prolonged air | ||||
leak); higher in | ||||
complicated cysts. | ||||
No late complications. | ||||
No mortality. | ||||
Recurrence in | ||||
de-epithelialised group | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Limaïauiem et al., | 33 patients from one | Presenting complaint | 94% symptomatic (chest | Management of all |
Lung, 2008, | centre over six years | pain, cough, | bronchogenic cysts | |
Tunisia, [20] | haemoptysis, dyspnoea, | based on complete | ||
Follow-up between | fever, dysphagia) | surgical excision. | ||
Retrospective | one and 51 months | Conclude that definitive | ||
cohort study (2b) | Preoperative | Correct diagnosis in | diagnosis is by histology | |
diagnosis | 33.3% | only and that complete | ||
surgical excision is | ||||
Postoperative | 14% Early complications | mandatory, although | ||
complications | (pneumothorax, | conclusions based on | ||
haemorrhage, pleural | findings of other studies | |||
effusion, seizure). | ||||
No late complications. | ||||
No mortality | ||||
Complications of | No patients | |||
conservative | conservatively managed | |||
management | ||||
Granato et al., | 30 adult patients treated | Presenting complaint | 30% symptomatic | Two symptomatic cysts |
Asian Cardiovasc | for bronchogenic cysts | (cough, sputum, pain, | complicated | |
Thorac Ann, 2009, | from one centre, over | fever, weakness) | intraoperatively by severe | |
Italy, [21] | 32 years | adhesions | ||
Preoperative | Correct diagnosis in | |||
Retrospective | diagnosis | 100% (CT or CT and | One case of large-cell | |
cohort study (2b) | MRI) | anaplastic carcinoma in | ||
wall of cyst | ||||
Postoperative | 10% intraoperative | |||
complications | complications. | Excision of | ||
10% postoperative | asymptomatic cysts | |||
complications | advocated to avoid | |||
complex surgery and | ||||
Complications of | No patients | complications, and also | ||
conservative | conservatively managed | to reduce malignant | ||
management | potential | |||
De Giacomo et al., | 30 adult patients from | Presenting complaint | 37% symptomatic | Authors feel that patients |
Eur J Cardiothorac Surg | one centre over | (cough, pain, dysphagia) | cannot be completely | |
2009, Italy, [22] | 12 years | assured about | ||
Postoperative | None | conservative | ||
Retrospective | Follow-up | complications | management but | |
cohort study (2b) | 3–120 months | acknowledge the | ||
Complications of | Asymptomatic patients | management is | ||
conservative | requested surgery | controversial | ||
management | because of enlarging | |||
cysts, risk of | ||||
complication or fear of | ||||
malignancy | ||||
Costa Júnior Ada | 60 patients with | Presenting complaint | 92% symptomatic | Heterogenous group of |
et al., J Bras | pulmonary | (cough, dyspnoea, pain, | patients and disorders. | |
Pneumol, 2008, | malformations | infection) | Difficult to extract | |
Brazil, [23] | (including 27 with | bronchogenic cyst data | ||
bronchogenic cyst) | Preoperative | ‘Frequent’ misdiagnosis | ||
Retrospective | from one centre | diagnosis | Prognosis noted to be | |
cohort study (2b) | over 35 years, | unpredictable. One | ||
including 40 paediatric | Postoperative | 23% (pneumonia, | patient found to have | |
patients | complications | atelectasis, empyema, | adenocarcinoma in wall | |
sepsis). | of cyst | |||
3.3% mortality | ||||
Complications of | Diagnosis/treatment | |||
conservative | delayed in three patients | |||
management | up to 36 months (mean | |||
15): outcome in these | ||||
patients not specified | ||||
Weber et al., | 12 patients from a | Presenting complaint | 42% symptomatic | Agree that management |
Ann Thorac Surg, | single centre | (cough, pain, pneumonia) | of asymptomatic cysts is | |
2004, Switzerland, | undergoing | controversial but that | ||
[24] | video-assisted | Preoperative | 100% correct diagnosis | there appears to be no |
thoracoscopic surgery | diagnosis | with CT with or without | need for urgent surgery | |
Retrospective | for bronchogenic | MRI. MRI noted to be | in these cases provided | |
cohort study (2b) | cysts, over seven years | superior | that a simple cyst has | |
been clearly diagnosed | ||||
40.5-month mean | Postoperative | None | ||
follow-up | complications | |||
Complications of | Six patients observed for | |||
conservative | between two and | |||
management | 22 years without | |||
complications.Three | ||||
developed mild | ||||
symptoms. | ||||
Three patients eventually | ||||
requested surgery | ||||
because of fear of | ||||
malignancy/complications | ||||
or enlarging cyst |
CT, computed tomography; EUS, endoscopic ultrasound; USS, ultrasound scan; FNA, fine needle aspiration.
6. Results
The prevalence rates for bronchogenic cysts are not known, with this rare condition thought to remain largely undiagnosed in an asymptomatic population. From the papers reviewed, the incidence was, on average, approximately 30 cases per institution (population sizes unknown) over a 20-year period (approx. 11–55). Some cysts were found incidentally (6–79%) but the remainder presented with a variety of symptoms including chest pain, dyspnoea, haemoptysis and recurrent chest infections. Rarely, the presentation was of a serious complication of the cysts, such as sepsis or compressive symptoms.
In total, the papers identified for this systematic review included 683 adult patients with bronchogenic cysts of whom 74 were either treated conservatively, or had diagnosis and/or treatment delayed up to 22 years. Of those treated conservatively, 31 (45%) subsequently developed symptoms and proceeded to surgical treatment. A number of studies determined that complicated bronchogenic cysts (for example ruptured or infected at the time of surgery) were associated with intraoperative difficulties. Only one study [10] compared the frequency of postoperative complications between patients who were symptomatic or asymptomatic preoperatively (27% and 14%, respectively), and found that these were not significantly different (P=0.596). There was no evidence in any of the reviewed papers that prolonged observation increased the incidence of complications.
Historically, it has been appropriate to operate on these asymptomatic bronchogenic cysts for both diagnostic and therapeutic purposes and the majority of the studies cited did proceed to operate on patients, both with and without symptoms. Ponn [25] commented on this dogmatic approach to tradition, making reference to a number of the studies described here [9, 12, 16]. With inclusion of only surgical cases in several of the smaller studies, the data presented is likely to be skewed.
Bronchogenic or benign cyst was diagnosed preoperatively in 50–100% of cases. Several studies made reference to the improvement in imaging over the last two decades, in particular to the benefits of magnetic resonance. Patients investigated in the latter part of studies were often noted to have more accurate preoperative diagnosis than those investigated 20 years earlier. Kanemitsu et al. [4] scanned all patients with MRI and reported 100% success rate in preoperative diagnosis, while Granato et al. [21] and Weber et al. [24] also commented on the superior diagnostic ability of MRI. In studies in which computerised tomography was used with or without other diagnostic procedures including ultrasound and mediastinoscopy, the diagnosis was made preoperatively with varying degrees of success.
Postoperative complications in the studies ranged from 0 to 27%. In patients who did suffer complications of surgery, the majority were generic operative complications with two cases attributed directly to cysts. In five patients (0.7% of all those studied), bronchogenic cysts were associated with malignancies: one squamous cell, one adenocarcinoma, two bronchoalveolar carcinomas and one large-cell anaplastic carcinoma. It was not stated whether these were due to malignant transformation or incidental finding, but the malignancies were usually found in the cyst wall.
Surgical excision of bronchogenic cysts has historically been performed for three main reasons:
to confirm diagnosis;
to prevent development of symptoms and/or complications and to pre-empt the possibility of surgery on complex inflammatory lesions;
to avoid any potential for malignant transformation.
Advances in non-invasive diagnostic techniques, in particular the role of MRI, may have rendered the first reason obsolete.
Nonetheless, the literature available demonstrates that the traditional approach of surgical treatment of all bronchogenic cysts gives acceptable results with low rates of recurrence, morbidity and postoperative mortality. Approximately half of patients who are asymptomatic at presentation will eventually go on to develop symptoms and/or complications, with a body of evidence which suggests that complications make surgery more challenging. No studies, however, have demonstrated that this is associated with a worse postoperative outcome than that of elective procedures.
Because of these concerns, only a very small proportion of the patients studied were followed up with conservative management instead of surgical excision (74/683).
7. Clinical bottom line
While there is good evidence to support the excision of symptomatic cysts, the evidence for conservative management of asymptomatic bronchogenic cysts is very limited, both because the majority of bronchogenic cysts present with bothersome symptoms and because conventional treatment has tended towards surgical management. Concerns that these benign entities may develop complications are not unfounded as 45% of asymptomatic patients studied here went on to develop symptoms. However, the success, morbidity and mortality of surgery are no different if operated on whilst asymptomatic or once complications have arisen. The other major concern – of missed malignancy – should be weighed in balance of its 0.7% risk, compared with the approximately 20% morbidity of surgery. Many surgeons have and continue to recommend surgery to asymptomatic adults with bronchogenic cysts and the literature would suggest this to be an acceptable approach. However, conservative management is a potentially viable alternative if close follow-up is possible.