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Spontaneous extracapsular parathyroid adenoma hemorrhage: when surgery is required?

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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

  1. 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

    Article  PubMed  PubMed Central  Google Scholar 

  2. 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

    Article  Google Scholar 

  3. 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

    Article  Google Scholar 

  4. 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

    Article  PubMed  PubMed Central  Google Scholar 

  5. 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

    Article  PubMed  Google Scholar 

  6. R.B. Capps, Multiple parathyroid tumors with massive mediastinal and subcutaneous hemorrhage. Am. J. Med Sci. 188, 801–4 (1934)

    Article  Google Scholar 

  7. 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

    Article  PubMed  PubMed Central  Google Scholar 

  8. 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

    Article  CAS  PubMed  Google Scholar 

  9. 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

    CAS  PubMed  PubMed Central  Google Scholar 

  10. 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

    Article  PubMed  Google Scholar 

  11. 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

    Article  Google Scholar 

  12. 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

    Article  PubMed  Google Scholar 

  13. 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

    Article  PubMed  Google Scholar 

  14. 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

    Article  Google Scholar 

  15. 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

    Article  CAS  PubMed  Google Scholar 

  16. 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

    Article  PubMed  Google Scholar 

  17. 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

    Article  PubMed  PubMed Central  Google Scholar 

  18. 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

    Article  PubMed  PubMed Central  Google Scholar 

  19. 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

    Article  PubMed  PubMed Central  Google Scholar 

  20. 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

    Article  CAS  PubMed  Google Scholar 

  21. 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

    Article  CAS  PubMed  Google Scholar 

  22. 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

    Article  PubMed  PubMed Central  Google Scholar 

  23. 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

    Article  CAS  Google Scholar 

  24. 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

    Article  PubMed  Google Scholar 

  25. 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

    Article  Google Scholar 

  26. 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

    Article  PubMed  PubMed Central  Google Scholar 

  27. 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

    Article  CAS  PubMed  Google Scholar 

  28. 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

    Article  CAS  PubMed  Google Scholar 

  29. 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

    Article  CAS  PubMed  Google Scholar 

  30. 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

    Article  CAS  PubMed  Google Scholar 

  31. 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

    Article  PubMed  Google Scholar 

  32. 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

    Article  Google Scholar 

  33. 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

    Article  CAS  PubMed  Google Scholar 

  34. 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

    Article  PubMed  Google Scholar 

  35. 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

    Article  PubMed  Google Scholar 

  36. 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

    Article  PubMed  PubMed Central  Google Scholar 

  37. 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

    Article  PubMed  Google Scholar 

  38. 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

    Article  CAS  PubMed  Google Scholar 

  39. 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)

    Article  Google Scholar 

  40. 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

    Article  PubMed  Google Scholar 

  41. 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

    Article  Google Scholar 

  42. 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

    Article  Google Scholar 

  43. 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

    Article  CAS  PubMed  Google Scholar 

  44. 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)

    Article  CAS  Google Scholar 

  45. 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

    Article  CAS  Google Scholar 

  46. 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)

    PubMed  Google Scholar 

  47. 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

    Article  PubMed  PubMed Central  Google Scholar 

  48. 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

    Article  CAS  PubMed  Google Scholar 

  49. 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

    Article  PubMed  PubMed Central  Google Scholar 

  50. 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

    Article  Google Scholar 

  51. 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

    Article  PubMed  Google Scholar 

  52. 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

    Article  PubMed  Google Scholar 

  53. 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

    Article  CAS  PubMed  Google Scholar 

  54. 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

    Article  CAS  Google Scholar 

  55. 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

    Article  PubMed  PubMed Central  Google Scholar 

  56. 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

    Article  PubMed  Google Scholar 

  57. 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

    Article  PubMed  Google Scholar 

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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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Correspondence to Marco Familiari.

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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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