Abstract
Purpose
Spontaneous bleeding is a rare but serious complication of parathyroid adenomas and few cases were reported in the literature. Clinical manifestations and treatment may vary but sometimes an immediate surgery is required. In other cases a conservative approach can be preferred based on clinical stability and patients’ conditions. The purpose of this work is to describe our case and to carry out a review of the current literature on this topic.
Methods
We reported a case of a parathyroid adenoma hemorrhage in an elderly patient describing its management. Moreover, a literature review of 57 cases was carried out, with the aim of collecting data about the most involved parathyroid gland and identifying the most correct management based on clinical manifestations and chosen treatments.
Results
The patients had an age between 29 and 81 years (mean 56.9 ± 16.4 years). Forty-four patients were females (77.2%), whereas 12 were males (21.8%). Based on clinical severity, age, comorbidities and calcemic status, three possible scenarios were identified, each with a recommended management.
Conclusions
In parathyroid adenoma hemorrhages a careful clinical assessment is crucial to identify emergency conditions requiring immediate intubation, tracheostomy or neck exploration. Elderlies and comorbid patients have a higher risk of perioperative complications and indication for surgery should be evaluated case by case: whenever feasible, a conservative approach should be preferred in these subjects, especially in those with a stable course and without hypercalcemia-related symptoms.
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References
J.J. van den Broek, M.M. Poelman, B.M. Wiarda, H.J. Bonjer, A.P. Houdijk, Extensive cervicomediastinal hematoma due to spontaneous hemorrhage of a parathyroid adenoma: a case report. J. Surg. Case Rep. 2015(5), rjv039 (2015). https://doi.org/10.1093/jscr/rjv039. Published 2015 May 1
M. Tonerini, E. Orsitto, L. Fratini et al. Cervical and mediastinal hematoma: presentation of an asymptomatic cervical parathyroid adenoma: case report and literature review. Emerg. Radio. 10(4), 213–215 (2004). https://doi.org/10.1007/s10140-003-0317-0
A.P. Devalapalli, D.T. Kashiwagi, Perioperative care of geriatric patients. Hosp. Pr. (1995) 48(sup1), 26–36 (2020). https://doi.org/10.1080/21548331.2020.1719713
L.S. Nidadavolu, A.L. Ehrlich, F.E. Sieber, E.S. Oh, Preoperative Evaluation of the Frail Patient. Anesth. Analg. 130(6), 1493–1503 (2020). https://doi.org/10.1213/ANE.0000000000004735
M.B. Hamel, W.G. Henderson, S.F. Khuri, J. Daley, Surgical outcomes for patients aged 80 and older: morbidity and mortality from major noncardiac surgery. J. Am. Geriatr. Soc. 53(3), 424–429 (2005). https://doi.org/10.1111/j.1532-5415.2005.53159.x
R.B. Capps, Multiple parathyroid tumors with massive mediastinal and subcutaneous hemorrhage. Am. J. Med Sci. 188, 801–4 (1934)
H. Shinomiya, N. Otsuki, S. Takahara et al. Parathyroid adenoma causing spontaneous cervical hematoma: two case reports. BMC Res. Notes 8, 726 (2015). https://doi.org/10.1186/s13104-015-1611-0. Published 2015 Nov 26
K.J. Simcic, M.T. McDermott, G.J. Crawford, W.H. Marx, J.L. Ownbey, G.S. Kidd, Massive extracapsular hemorrhage from a parathyroid cyst. Arch. Surg. 124(11), 1347–1350 (1989). https://doi.org/10.1001/archsurg.1989.01410110109023
C. Zhao, X. Wang, H. Wei, G. Ma, Parathyroid adenoma causing a spontaneous cervical and mediastinal massive hematoma. Int J. Clin. Exp. Med. 8(11), 21826–21829 (2015). Published 2015 Nov 15
P.C. Chaffanjon, N. Chavanis, O. Chabre, P.Y. Brichon, Extracapsular hematoma of the parathyroid glands. World J. Surg. 27(1), 14–17 (2003). https://doi.org/10.1007/s00268-002-6429-y
A. Martínez-Martínez, J. García-Espinosa, Cervical haematoma secondary to spontaneous bleeding of parathyroid adenoma. Hematoma cervical por sangrado espontáneo de adenoma paratiroideo. Acta Otorrinolaringol. Esp. 70(1), 59–60 (2019). https://doi.org/10.1016/j.otorri.2018.01.001
I. Tessler, M. Adi, J. Diment, Y. Lahav, D. Halperin, O. Cohen, Spontaneous neck hematoma secondary to parathyroid adenoma: a case series [published correction appears in Eur Arch Otorhinolaryngol. 2020 Apr 29;:]. Eur. Arch. Otorhinolaryngol. 277(9), 2551–2558 (2020). https://doi.org/10.1007/s00405-020-05959-z
L. An, T. Ji, L. Li, Diffuse hematoma caused by spontaneous rupture of a parathyroid adenoma: a case report. Braz. J. Otorhinolaryngol. 86(Suppl 1), 48–50 (2020). https://doi.org/10.1016/j.bjorl.2017.06.006
S. Khan, C.C. Choe, A. Shabaik, M. Bouvet, Parathyroid adenoma presenting with spontaneous cervical and anterior mediastinal hemorrhage: a case report. Med. (Baltim.) 98(5), e14347 (2019). https://doi.org/10.1097/MD.0000000000014347
M.C. Zillikens, A. Wijbenga, Images in clinical medicine. Extracapsular hemorrhage from a parathyroid adenoma. N. Engl. J. Med. 359(11), 1155 (2008). https://doi.org/10.1056/NEJMicm075840
N. Yoshimura, H. Mukaida, T. Mimura et al. A case of an acute cervicomediastinal hematoma secondary to the spontaneous rupture of a parathyroid adenoma. Ann. Thorac. Cardiovasc Surg. 20(Suppl), 816–820 (2014). https://doi.org/10.5761/atcs.cr.12.02060
B.A. Whitson, R. Heckmann, C. Manivel, A. Anne Lassig, J. D’Cunha, Acute airway compromise from a hemorrhagic posterior cervical-mediastinal mass: Rare presentation of a parathyroid adenoma. J. Thorac. Dis. 3(1), 68–70 (2011). https://doi.org/10.3978/j.issn.2072-1439.2010.11.7
L. Ulrich, G. Knee, C. Todd, Spontaneous cervical haemorrhage of a parathyroid adenoma. Endocrinol. Diabetes Metab. Case Rep. 2015, 150034 (2015). https://doi.org/10.1530/EDM-15-0034
W.S. Shim, I.K. Kim, S.D. Yoo, D.H. Kim, Non-functional parathyroid adenoma presenting as a massive cervical hematoma: a case report. Clin. Exp. Otorhinolaryngol. 1(1), 46–48 (2008). https://doi.org/10.3342/ceo.2008.1.1.46
G.H. Santos, C.L. Tseng, R.W. Frater, Ruptured intrathoracic parathyroid adenoma. Chest 68(6), 844–846 (1975). https://doi.org/10.1378/chest.68.6.844
N. Santelmo, S. Hirschi, D. Sadoun et al. Bilateral hemothorax revealing mediastinal parathyroid adenoma. Respiration 66(2), 176–178 (1999). https://doi.org/10.1159/000029364
H.U. Rehman, M. Markovski, A. Khalifa, Spontaneous cervical hematoma associated with parathyroid adenoma. CMAJ 182(13), E632 (2010). https://doi.org/10.1503/cmaj.091167
A. Paracchi, E. Reschini, C. Ferrari, R. Macchi, Asymptomatic parathyroid adenoma manifested by intratumoral hemorrhage. J. Endocrinol. Investig. 13(8), 667–670 (1990). https://doi.org/10.1007/BF03349591
I. Osorio Silla, L. Lorente, J.J. Sancho, A. Sitges-Serra, Spontaneous neck hematoma due to rupture of parathyroid adenoma: report of 3 cases and literature review. Endocrinol. Nutr. 61(1), e5–e6 (2014). https://doi.org/10.1016/j.endonu.2013.04.014
T. Nito, C. Miyajima, M. Kimura, M. Sugasawa, Parathyroid adenoma causing spontaneous cervical hematoma: a case report. Acta Otolaryngol. Suppl. 559, 160–163 (2007). https://doi.org/10.1080/03655230701600491
I. Merante-Boschin, M. Fassan, M.R. Pelizzo, E.C. Ide, M. Rugge, Neck emergency due to parathyroid adenoma bleeding: a case report. J. Med. Case Rep. 3, 7404 (2009). https://doi.org/10.1186/1752-1947-3-7404. Published 2009 May 21
S. Maweja, F. Sebag, J. Hubbard, C. Misso, J.F. Henry, Hématome cervical spontané secondaire à une hémorragie extracapsulaire d’un adénome parathyroïdien: à propos de 2 cas [Spontaneous cervical haematoma due to extracapsular haemorrhage of a parathyroid adenoma: a report of 2 cases]. Ann. Chir. 128(8), 561–562 (2003). https://doi.org/10.1016/s0003-3944(03)00184-6
P. Ku, P. Scott, J. Kew, A. van Hasselt, Spontaneous retropharyngeal haematoma in a parathyroid adenoma. Aust. N. Z. J. Surg. 68(8), 619–621 (1998). https://doi.org/10.1111/j.1445-2197.1998.tb02117.x
A.M. Korkis, P.F. Miskovitz, Acute pharyngoesophageal dysphagia secondary to spontaneous hemorrhage of a parathyroid adenoma. Dysphagia 8(1), 7–10 (1993). https://doi.org/10.1007/BF01351471
M. Kihara, H. Yokomise, A. Yamauchi, A. Irie, K. Matsusaka, A. Miyauchi, Spontaneous rupture of a parathyroid adenoma presenting as a massive cervical hemorrhage: report of a case. Surg. Today 31(3), 222–224 (2001). https://doi.org/10.1007/s005950170172
E. Ilyicheva, Spontaneous cervical-mediastinal hematoma caused by hemorrhage into parathyroid adenoma: a clinical case. Int J. Surg. Case Rep. 6C, 214–217 (2015). https://doi.org/10.1016/j.ijscr.2014.10.029
J. Huang, A. Soskos, S.M. Murad, B.R. Krawisz, S.H. Yale, A.C. Urquhart, Spontaneous hemorrhage of a parathyroid adenoma into the mediastinum. Endocr. Pr. 18(4), e57–e60 (2012). https://doi.org/10.4158/EP11329.CR
W.P. Hellier, A. McCombe, Extracapsular haemorrhage from a parathyroid adenoma presenting as a massive cervical haematoma. J. Laryngol. Otol. 111(6), 585–587 (1997). https://doi.org/10.1017/s0022215100137995
M.A. Gorris, M.V. Guido, M.S. Rawla, J.A. Aloi, Parathyroid Hemorrhage. J. Emerg. Med. 52(5), e193–e194 (2017). https://doi.org/10.1016/j.jemermed.2017.01.023
D.J. Givens, J.P. Hunt, B.G. Bentz, Uncommon presentations of parathyroid adenoma. Head. Neck 35(9), E265–E268 (2013). https://doi.org/10.1002/hed.23124
A. Garrahy, D. Hogan, J.P. O’Neill, A. Agha, Acute airway compromise due to parathyroid tumour apoplexy: an exceptionally rare and potentially life-threatening presentation [published correction appears in BMC Endocr Disord. 2017 Jul 14;17 (1):42]. BMC Endocr. Disord. 17(1), 35 (2017). https://doi.org/10.1186/s12902-017-0186-2. Published 2017 Jun 21
A. Devèze, F. Sebag, S. Pili, J.F. Henry, Parathyroid adenoma disclosed by a massive cervical hematoma. Otolaryngol. Head. Neck Surg. 134(4), 710–712 (2006). https://doi.org/10.1016/j.otohns.2005.03.075
B.E. Berry, P.C. Carpenter, R.E. Fulton, G.K. Danielson, Mediastinal hemorrhage from parathyroid adenoma simulating dissecting aneurysm. Arch. Surg. 108(5), 740–741 (1974). https://doi.org/10.1001/archsurg.1974.01350290102019
G. Koulouris, M. Pianta, S. Stuckey, The ‘sentinel clot’ sign in spontaneous retropharyngeal hematoma secondary to parathyroid apoplexy. Ear Nose Throat J. 85(9), 606–608 (2006)
Y. Shundo, H. Nogimura, Y. Kita, H. Neyatani, R. Kobayashi, H. Sugimura, Spontaneous parathyroid adenoma hemorrhage. Jpn J. Thorac. Cardiovasc. Surg. 50(9), 391–394 (2002). https://doi.org/10.1007/BF02913192
J.Y. Choi, H.J. Yoo, Y.H. Park, S.H. Cho, A Case of Spontaneous Extracapsular Hemorrhage of Intrathyroidal Parathyroid Adenoma. Korean J. Otorhinolaryngol.-Head. Neck Surg. 53, 245–7 (2010). https://doi.org/10.3342/kjorl-hns.2010.53.4.245
T.H. Shin, S.S. Park, C.S. Won, M.K. Kim, M.S. Kim, Parathyroid Adenoma Causing Spontaneous Cervical Hematoma: a Case Report and Review of Literature. Korean J. Head. Neck Oncol. 35(2 Nov), 27–30 (2019). https://doi.org/10.21593/kjhno/2019.35.2.27
L.S. Hotes, J. Barzilay, L.P. Cloud, A.R. Rolla, Spontaneous hematoma of a parathyroid adenoma. Am. J. Med. Sci. 297(5), 331–333 (1989). https://doi.org/10.1097/00000441-198905000-00012
Y. Amano, I. Fukuda, H. Mori, T. Kumoi, Hemorrhage from spontaneous rupture of a parathyroid adenoma (a case report). Ear Nose Throat J. 72(12), 794–799 (1993)
W. Kozlow, M.J. Demeure, L.M. Welniak, J.L. Shaker, Acute extracapsular parathyroid hemorrhage: case report and review of the literature. Endocr. Pr. 7(1), 32–36 (2001). https://doi.org/10.4158/EP.7.1.32
M.V. Bürgueser, A. Diller, M.E. Bustos, D.M. Debernardi, F. Bernabeu, Hematoma mediastinal espontáneo secundario a ruptura de adenoma quístico de paratiroides ectópica [Spontaneous mediastinal hematoma as initial presentation of cystic adenoma of ectopic parathyroid]. Rev. Fac. Cien Med. Univ. Nac. Cordoba 68(4), 164–168 (2011)
P. Carneiro de Sousa, I. Gambôa, D. Duarte, N. Trigueiros-Cunha, Nontraumatic Parapharyngeal Haematoma: a Rare Lesion. Case Rep. Otolaryngol. 2018, 7340937 (2018). https://doi.org/10.1155/2018/7340937. Published 2018 Oct 24
S.H. Sun, J. Zeng, Y. Peng, Y. Song, W.N. Hu, F.R. Ma, Zhonghua Er Bi Yan Hou Tou Jing Wai Ke Za Zhi 54(11), 865–867 (2019). https://doi.org/10.3760/cma.j.issn.1673-0860.2019.11.012
N. Nakatake, S. Matsubayashi, T. Hara, S. Satoh, H. Yamashita, Primary Hyperparathyroidism Accompanied by Recovery of Parathyroid Blood Flow Three Months After Spontaneous Parathyroid Hemorrhage. AACE Clin. Case Rep. 6(6), e322–e325 (2020). https://doi.org/10.4158/ACCR-2020-0311. Published 2020 Sep 24
NIH conference, Diagnosis and management of asymptomatic primary hyperparathyroidism: consensus development conference statement. Ann. Intern Med. 114(7), 593–597 (1991). https://doi.org/10.7326/0003-4819-114-7-593
F.E. Turrentine, H. Wang, V.B. Simpson, R.S. Jones, Surgical risk factors, morbidity, and mortality in elderly patients. J. Am. Coll. Surg. 203(6), 865–877 (2006). https://doi.org/10.1016/j.jamcollsurg.2006.08.026
C.T. Wootten, E.A. Orzeck, Spontaneous remission of primary hyperparathyroidism: a case report and meta-analysis of the literature. Head. Neck 28(1), 81–88 (2006). https://doi.org/10.1002/hed.20316
E. Nylen, A. Shah, J. Hall, Spontaneous remission of primary hyperparathyroidism from parathyroid apoplexy. J. Clin. Endocrinol. Metab. 81(4), 1326–1328 (1996). https://doi.org/10.1210/jcem.81.4.8636326
F. Cetani, E. Ambrogini, P. Faviana et al. Spontaneous short-term remission of primary hyperparathyroidism from infarction of a parathyroid adenoma. J. Endocrinol. Investig. 27(7), 687–690 (2004). https://doi.org/10.1007/BF03347505
E.I. Efremidou, M.S. Papageorgiou, E. Pavlidou, K.J. Manolas, N. Liratzopoulos, Parathyroid apoplexy, the explanation of spontaneous remission of primary hyperparathyroidism: a case report. Cases J. 2, 6399 (2009). https://doi.org/10.1186/1757-1626-2-6399. Published 2009 Mar 10
H. Boyd-Carson, T. Gana, S. Lockwood, D. Murray, G.M. Tierney, A review of surgical and peri-operative factors to consider in emergency laparotomy care. Anaesthesia 75(Suppl 1), e75–e82 (2020). https://doi.org/10.1111/anae.14821
H. Cheng, J.W. Clymer, B. Po-Han Chen et al. Prolonged operative duration is associated with complications: a systematic review and meta-analysis. J. Surg. Res. 229, 134–144 (2018). https://doi.org/10.1016/j.jss.2018.03.022
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All authors contributed to the study conception and design. Material preparation, data collection and analysis were performed by M.F., D.D.S., A.G., G.D., L.G. and R.M. The first draft of the paper was written by M.F., D.D.S. and A.G. and all authors commented on previous versions of the paper. Tables were made by M.F., A.G. and G.D. The supervision was performed by M.B. and L.G. All authors read and approved the final paper.
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Familiari, M., Di Santo, D., Galli, A. et al. Spontaneous extracapsular parathyroid adenoma hemorrhage: when surgery is required?. Endocrine 75, 575–582 (2022). https://doi.org/10.1007/s12020-021-02876-x
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DOI: https://doi.org/10.1007/s12020-021-02876-x