Year 2021 Vol. 29 No 5

CASE REPORTS

S.A. PLAKSIN

CYSTIC PULMONARY HAMARTOMA MANIFESTING AS SPONTANEOUS PNEUMOTHORAX

E.A. Vagner Perm State Medical University, Perm,
The Russian Federation

Cystic hamartoma refers to rare benign lung tumors. The literature describes 17 cases of this disease. The tumor may look like multiple bilateral cysts or a solitary single-chamber or multi-chamber cyst of a large size, which makes it difficult to diagnose. The disease can be complicated by spontaneous pneumothorax. The cyst itself often looks like a pneumothorax. An observation of a 52-year-old man complaining of chest pain and shortness of breath is presents. On the x-ray, the left lung is compressed with air, which is regarded as a spontaneous pneumothorax. Two-day drainage did not give any results. The video-assisted thoracoscopic surgery technique was performed and a large air cyst was detected. A conversion to a thoracotomy was made. A cyst of 20×15 cm size originated from the lower lobe, the lung was in atelectasis. Cyst resection and lung decortication were performed. Histological examination of the cyst wall revealed a hamartoma of the lung. The postoperative period was uneventful. Differential diagnosis of cystic hamartoma is conducting with lymphangioleiomyomatosis, air cysts, extrapulmonary sequestration, echinococcal cysts, and lung cancer. Indications for surgery are the following: chest pain, shortness of breath, pneumothorax, and hemoptysis. In a unilateral process, a cyst resection or lobectomy have to be performed. Cystic pulmonary hamartoma should be included in the differential diagnostic range in patients with recurrent spontaneous pneumothorax, hemoptysis, single and multiple lung cysts. It is impossible to determine the diagnosis without a biopsy and histological examination.

Keywords: cystic lung hamartoma, spontaneous pneumothorax, air lung cyst, benign lung tumors, videothoracoscopy
p. 624-628 of the original issue
References
  1. Shields TW, Locicero IIIJ, Reed CE, Feins RH, eds. General thoracic surgery. Wolters Kluwer/Lippincott Williams and Wilkins; 2009. 2616 p.
  2. Kuzdzal J, ed. ESTS Textbook of thoracic surgery. Cracow: medycyna praktyczna; 2014. 1084 p.
  3. Sellke FW, del Nido PJ, Swanson SJ, eds. 9th ed. Elsevier; 2016. 2586 p.
  4. Abu Omar M, Abu Ghanimeh M, Tylski E, Saettele T. Endobronchial hamartoma: a rare disease with more common presentation. BMJ Case Rep. 2016 Nov 7;2016:bcr2016216771. doi: 10.1136/bcr-2016-216771
  5. Fasanya AA, Hattab Y, Patel A, Lega M. Mesenchymal cystic hamartoma of the lung. Respir Med Case Rep. 2017 May 12;21:158-60. doi: 10.1016/j.rmcr.2017.05.004. eCollection 2017.
  6. van Klaveren RJ, Hassing HH, Wiersma-van Tilburg JM, Lacquet LK, Cox AL. Mesenchymal cystic hamartoma of the lung: a rare cause of relapsing pneumothorax. Thorax. 1994 Nov;49(11):1175-76. doi: 10.1136/thx.49.11.1175
  7. Chadwick SL, Corrin B, Hansell DM, Geddes DM. Fatal haemorrhage from mesenchymal cystic hamartoma of the lung. Eur Respir J. 1995 Dec;8(12):2182-84. doi: 10.1183/09031936.95.08122182
  8. Saadi MM, Barakeh DH, Husain S, Hajjar WM. Large multicystic pulmonary chondroid hamartoma in a child presenting as pneumothorax. Saudi Med J. 2015 Apr;36(4):487-89. doi: 10.15537/smj.2015.4.10210
  9. Chida M, Minowa M, Eba S, Odajima H. Mesenchymal cystic hamartoma of the lung: a rare cause of pneumothorax. Gen Thorac Cardiovasc Surg. 2009 Mar;57(3):166-68. doi: 10.1007/s11748-008-0333-9
  10. Ozbudak IH, Dertsiz L, Bassorgun CI, Ozbilim G. Giant cystic chondroid hamartoma of the lung. J Pediatr Surg. 2008 Oct;43(10):1909-11. doi: 10.1016/j.jpedsurg.2008.06.007
  11. Yorita K, Ayabe T, Chosa E, Uchino N, Nagatomo Y, Yamaguchi T, Nakatani Y, Kataoka H. Unusual cystic hamartomatous lung lesion with clinical manifestation of subpleural bullae in a woman of reproductive age: A case report. Pathol Int. 2015 Oct;65(10):558-62. doi: 10.1111/pin.12342
  12. Kim GY, Han J, Kim DH, Kim J, Lee KS. Giant cystic chondroid hamartoma. J Korean Med Sci. 2005 Jun;20(3):509-11. doi: 10.3346/jkms.2005.20.3.509
  13. Argemi X, Santelmo N, Matau C, Croce S, Weingertner N. Cystic lung lesions mimicking hydatidosis: hamartoma or benign metastazing leiomyoma? Ann Thorac Surg. 2019 Sep;108(3):e195-e198. doi: 10.1016/j.athoracsur.2019.01.044
  14. Greenspon A, Samuels L, Greenspon L. Extralobar sequestration complicated by a cystic hamartoma in an adult. Ann Thorac Surg. 2019;108(1):e43-e44. doi: 10.1016/j.athoracsurg.2018.10.072
  15. Glezos J, Toppin D, Cooney T. Mesenchymal cystic hamartoma presenting with spontaneous pneumothorax. Can Respir J. 2003 Jul-Aug;10(5):280-81. doi: 10.1155/2003/595082
Address for correspondence:
614990, Russian Federation,
Perm, Petropavlovskaya Str., 26,
E.A. Vagner Perm State Medical University,
the Surgery Department with the Course
of Cardiovascular Surgery
and Interventional Cardiology
tel. +7 342 239-29-72,
e-mail: splaksin@mail.ru,
Plaksin Sergei A.
Information about the authors:
Plaksin Sergei A., MD, Professor of the Surgery Department with the Course of Cardiovascular Surgery and Interventional Cardiology
E.A.Vagner Perm State Medical University, Perm, Russian Federation.
http://orcid.org/0000-0001-8108-1655
Contacts | ©Vitebsk State Medical University, 2007-2023