Airway hemangioma: a case report and literature review_Chinese Journal of Respiratory and Critical Care Medicine

Authors：

LIU Ting , WANG Faping , WANG Geng ,  MAO Hui

Department of Respiratory and Critical Care Medicine, West China Hospital, Sichuan University, Chengdu, Sichuan 610041, P.R.China;

Corresponding?author：

MAO Hui, Email: merrymh@126.com

Keywords：

Hemangioma; Trachea; Bronchus; Diagnosis

DOI：

10.7507/1671-6205.201610001

Video：

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ObjectiveTo explore the clinical features, diagnosis, differential diagnosis, pathological characteristics, management and prognosis of airway hemangioma, and improve the level of the diagnosis and therapy.MethodsThe clinical data of a patient with airway hemangioma admitted to West China Hospital of Sichuan University were retrospectively analyzed. The related literatures were reviewed. Databases including PubMed, Ovid Medline, Embase, VIP, Wanfang and Chinese National Knowledge infrastructure were searched using the keywords as " Trachea” OR " Bronchus” AND " Hemangioma” from January 1976 to October 2016.ResultsThe patient was a 32-year-old male presented with hemoptysis and backache more than 10 days. His enhanced chest CT scan revealed thickening of the esophagus wall and narrowing of the lumen of esophagus. A wide range of vascular tumor like changes in the trachea and the two sides of bronchus were found by the fiberoptic bronchoscopy and gastroscope examination suggested esophageal varices. Although the patient had a wide range of airway lesions, the symptom of hemoptysis was relieved after the conservative treatment. The patient’s condition was stable until now. From January 1976 to October 2016, a total of 34 related articles were retrieved and 36 cases of airway hemangioma were reported. The etiology of the disease remains unknown. Recurrent hemoptysis was the common symptoms of airway hemangioma, and imaging changes lacking characteristics. Histopathological examination revealed dilated, thin-walled and different size of vascular or hyperplastic capillary and immunohistochemical staining was positive for vascular endothelial cell specific CD34 and Ⅷ factor.ConclusionsHemangioma is a common benign tumor of head and neck in infants and children. Hemangioma is very rarely occurred in trachea in adults. Clinical and imaging manifestations are not specific, and diagnosis relies on histopathological examination. Fiberoptic bronchoscopy and three dimensional reconstruction of thoracic vessels with CT play important roles in the whole process of diagnosis and treatment of airway hemangioma. It should be differentiated from lymphangioma. The prognosis of airway hemangioma is good.

Citation： LIU Ting, WANG Faping, WANG Geng, MAO Hui. Airway hemangioma: a case report and literature review. Chinese Journal of Respiratory and Critical Care Medicine, 2017, 16(4): 387-393. doi: 10.7507/1671-6205.201610001 Copy

1.	Werner JA, Dünne AA, Folz BJ, et al. Current concepts in the classification, diagnosis and treatment of hemangiomas and vascular malformations of the head and neck. Eur Arch Otorhinolaryngol, 2001, 258(3): 141-149.
2.	Dabó H, Gomes R, Teixeira N, et al. Tracheal lobular capillary hemangioma treated with laser photocoagulation. J Bras Pneumol, 2016, 42(1): 72-73.
3.	Acharya MN, Kotidis K, Loubani M, et al. Tracheal lobular capillary haemangioma: a rare benign cause of recurrent haemoptysis. Case Rep Surg, 2016, 2016: 6290424.
4.	Kundu S, Dhua A, Hariprasath K, et al. Isolated endobronchial capillary haemangioma: a rare cause of haemoptysis in adult. Indian J Chest Dis Allied Sci, 2015, 57(2): 109-111.
5.	Xu Q, Yin X, Sutedjo J, et al. Lobular capillary hemangioma of the trachea. Arch Iran Med, 2015, 18(2): 127-129.
6.	楊曉紅, 陳穎, 魏雪梅, 等. 氣管小葉毛細血管瘤一例. 中華結核和呼吸雜志, 2015, 38(11): 865-867.
7.	Yu Y, Lee S, An J, et al. Massive hemoptysis due to endotracheal hemangioma: a case report and literature review. Tuberc Respir Dis, 2015, 78(2): 106-111.
8.	Prakash S, Bihari S, Wiersema U. A rare case of rapidly enlarging tracheal lobular capillary hemangioma presenting as difficult to ventilate acute asthma during pregnancy. BMC Pulm Med, 2014, 14: 41.
9.	Cho NJ, Baek AR, Kim J, et al. A case of capillary hemangioma of lingular segmental bronchus in adult. Tuberc Respir Dis, 2013, 75(1): 36-39.
10.	Jennings S, Tharion J, Jones P, et al. Bronchial haemangioma: exceptionally rare cause of hemoptysis. Heart Lung Circ, 2013, 22(12): 1030-1032.
11.	陳乾坤, 汪浩, 包敏偉, 等. 氣管黏膜血管瘤 1 例. 中華胸心血管外科雜志, 2013, 29(9): 570.
12.	Amy FT, Enrique DG. Lobular capillary hemangioma in the posterior trachea: a rare cause of hemoptysis. Case Rep Pulmonol, 2012, 2012: 592524.
13.	Shen J, Liu HR, Zhang FQ. Brachytherapy for tracheal lobular capillary haemangioma (LCH). J Thorac Oncol, 2012, 7(5): 939-940.
14.	Udoji TN, Bechara RI. Pyogenic granuloma of the distal trachea: a case report. J Bronchology Interv Pulmonol, 2011, 18(3): 281-284.
15.	雷靜, 韓丹, 段慧. 氣管毛細血管瘤一例. 中華放射學雜志, 2011, 45(6): 604.
16.	Chawla M, Stone C, Simoff MJ. Lobular capillary hemangioma of the trachea the second case. J Bronchology Interv Pulmonol, 2010, 17(3): 238-240.
17.	Kim HJ, Bae SY, Sung YK, et al. A case of tracheal capillary hemangioma in an adult. Tuberc Respir Dis, 2010, 69(5): 385-388.
18.	韓志海, 馮華松, 李偉卿, 等. 經電子支氣管鏡氬等離子氣體凝固治療主氣管內血管瘤 1 例. 中國內鏡雜志, 2010, 16(6): 671-672.
19.	葛瑩, 伍建林, 張麗枝. 支氣管粘膜下血管瘤一例. 中華放射學雜志, 2010, 44(5): 558.
20.	劉芳蕾, 陳國恩, 周畔, 等. 氣管小葉毛細血管瘤二例并文獻復習. 中華呼吸和結核雜志, 2010, 33(11): 849-852.
21.	Rose AS, Mathur PN. Endobronchial capillary hemangioma: case report and review of the literature. Respiration, 2008, 76(2): 221-224.
22.	Bellia M, Lo Casto A, Guddo F, et al. A rare case of pedunculated bronchial hemangioma. Monaldi Arch Chest Dis, 2008, 69(4): 189-191.
23.	Porfyridis I, Zisis C, Glinos K, et al. Recurrent cough and hemoptysis associated with tracheal capillary hemangioma in an adolescent boy: a case report. J Thorac Cardiovasc Surg, 2007, 134(5): 1366-1367.
24.	Zhu L, Wang YQ, Li HC. Extirpation of bronchial hemangioma using bronchoscope: case report. Chin Med J, 2006, 119(3): 259-266.
25.	Cordos I, Ulmeanu R, Mihaltan FD, et al. Sequential approach in a case of tracheal hemangioma: segmental tracheal resection after Nd-YAG laser application. Pneumologia, 2005, 54(4): 191-194.
26.	Madhumita K, Sreekumar KP, Malini H, et al. Tracheal haemangioma: case report. J Laryngol Otol, 2004, 118(8): 655-658.
27.	Irani S, Brack T, Pfaltz M, et al. Tracheal lobular capillary hemangioma: a rare cause of recurrent hemoptysis. Chest, 2003, 123(6): 2148-2149.
28.	Zambudio AR, Calvo MJ, Lanzas JT, et al. Massive hemoptysis caused by tracheal hemangioma treated with interventional radiology. Ann Thorac Surg, 2003, 75(4): 1302-1304.
29.	余雪濤, 方偉強. 氣管內海綿狀血管瘤 1 例. 中國實用內科雜志, 2003, 23(5): 310.
30.	張遜, 姚計方, 張澤峰. 氣管血管瘤一例報告. 中國肺癌雜志, 2001, 4(2): 143.
31.	So SC, Kwack KK, Park HK, et al. A case of tracheal hemangioma manifested massive hemoptysis. Tuberc Respir Dis, 1999, 47(5): 704-708.
32.	Strausz J, Soltesz I. bronchial capillary hemangioma in adults. Pathol Oncol Res, 1999, 5(3): 233-234.
33.	Brown JS, Rebeiz EE. Pathologic quiz case 1. Capillary hemangioma. Arch Otolaryngol Head Neck Surg, 1993, 119(6): 700-702.
34.	Wigton RB, Rohatgi PK. Isolated bronchial capillary hemangioma: a rare benign cause of hemoptysis. South Med J, 1979, 72(10): 1339-1340.
35.	Harding JR, Williams J, Seal RM. Pedunculated capillary hemangioma of the bronchus. Br J Dis Chest, 1978, 72(4): 336-342.
36.	馬蕓, 馬利君, 趙麗敏. 氣道毛細血管瘤病例報告及文獻復習. 中國內鏡雜志, 2014, 20(10): 1116-1118.
37.	Sweetser TH. Hemangioma of the larynx. Laryngoscope, 2009, 31(10): 797-806.
38.	Ferretti GR, Bithigofer C, Righini CA, et al. Imaging of tumors of the trachea and central bronchi. Radiol Clin North Am, 2009, 47(2): 227-241.
39.	林一丹, 蔣光亮. 胸部血管瘤的臨床特點與外科治療. 中國胸心血管外科臨床雜志, 2013, 20(1): 67-69.
40.	于濤. 血管瘤發病機制的研究進展. 國際口腔醫學雜志, 2007, 34(2): 122-124.
41.	Yoshino N, Okada D, Ujiie H, et al. Venous hemangioma of the posterior mediastinum. Ann Thorac Cardiovasc Surg, 2012, 18(3): 247-250.
42.	Schneider T, Storz K, Dienemann H, et al. Management of iatrogenic tracheobronchial injuries: a retrospective analysis of 29 cases. Ann Thorac Surg, 2007, 83(6): 1960-1964.
43.	Vivas-Colmenares GV, Fernandez-Pineda I, Lopez-Gutierrez JC, et al. Analysis of the therapeutic evolution in the management of airway infantile hemangioma. World J Clin Pediatr, 2016, 5(1): 95-101.
44.	Sakr L, Dutau H. Massive hemoptysis: an update on the role of bronchoscopy in diagnosis and management. Respiration, 2010, 80(1): 38-58.

1. Werner JA, Dünne AA, Folz BJ, et al. Current concepts in the classification, diagnosis and treatment of hemangiomas and vascular malformations of the head and neck. Eur Arch Otorhinolaryngol, 2001, 258(3): 141-149.
2. Dabó H, Gomes R, Teixeira N, et al. Tracheal lobular capillary hemangioma treated with laser photocoagulation. J Bras Pneumol, 2016, 42(1): 72-73.
3. Acharya MN, Kotidis K, Loubani M, et al. Tracheal lobular capillary haemangioma: a rare benign cause of recurrent haemoptysis. Case Rep Surg, 2016, 2016: 6290424.
4. Kundu S, Dhua A, Hariprasath K, et al. Isolated endobronchial capillary haemangioma: a rare cause of haemoptysis in adult. Indian J Chest Dis Allied Sci, 2015, 57(2): 109-111.
5. Xu Q, Yin X, Sutedjo J, et al. Lobular capillary hemangioma of the trachea. Arch Iran Med, 2015, 18(2): 127-129.
6. 楊曉紅, 陳穎, 魏雪梅, 等. 氣管小葉毛細血管瘤一例. 中華結核和呼吸雜志, 2015, 38(11): 865-867.
7. Yu Y, Lee S, An J, et al. Massive hemoptysis due to endotracheal hemangioma: a case report and literature review. Tuberc Respir Dis, 2015, 78(2): 106-111.
8. Prakash S, Bihari S, Wiersema U. A rare case of rapidly enlarging tracheal lobular capillary hemangioma presenting as difficult to ventilate acute asthma during pregnancy. BMC Pulm Med, 2014, 14: 41.
9. Cho NJ, Baek AR, Kim J, et al. A case of capillary hemangioma of lingular segmental bronchus in adult. Tuberc Respir Dis, 2013, 75(1): 36-39.
10. Jennings S, Tharion J, Jones P, et al. Bronchial haemangioma: exceptionally rare cause of hemoptysis. Heart Lung Circ, 2013, 22(12): 1030-1032.
11. 陳乾坤, 汪浩, 包敏偉, 等. 氣管黏膜血管瘤 1 例. 中華胸心血管外科雜志, 2013, 29(9): 570.
12. Amy FT, Enrique DG. Lobular capillary hemangioma in the posterior trachea: a rare cause of hemoptysis. Case Rep Pulmonol, 2012, 2012: 592524.
13. Shen J, Liu HR, Zhang FQ. Brachytherapy for tracheal lobular capillary haemangioma (LCH). J Thorac Oncol, 2012, 7(5): 939-940.
14. Udoji TN, Bechara RI. Pyogenic granuloma of the distal trachea: a case report. J Bronchology Interv Pulmonol, 2011, 18(3): 281-284.
15. 雷靜, 韓丹, 段慧. 氣管毛細血管瘤一例. 中華放射學雜志, 2011, 45(6): 604.
16. Chawla M, Stone C, Simoff MJ. Lobular capillary hemangioma of the trachea the second case. J Bronchology Interv Pulmonol, 2010, 17(3): 238-240.
17. Kim HJ, Bae SY, Sung YK, et al. A case of tracheal capillary hemangioma in an adult. Tuberc Respir Dis, 2010, 69(5): 385-388.
18. 韓志海, 馮華松, 李偉卿, 等. 經電子支氣管鏡氬等離子氣體凝固治療主氣管內血管瘤 1 例. 中國內鏡雜志, 2010, 16(6): 671-672.
19. 葛瑩, 伍建林, 張麗枝. 支氣管粘膜下血管瘤一例. 中華放射學雜志, 2010, 44(5): 558.
20. 劉芳蕾, 陳國恩, 周畔, 等. 氣管小葉毛細血管瘤二例并文獻復習. 中華呼吸和結核雜志, 2010, 33(11): 849-852.
21. Rose AS, Mathur PN. Endobronchial capillary hemangioma: case report and review of the literature. Respiration, 2008, 76(2): 221-224.
22. Bellia M, Lo Casto A, Guddo F, et al. A rare case of pedunculated bronchial hemangioma. Monaldi Arch Chest Dis, 2008, 69(4): 189-191.
23. Porfyridis I, Zisis C, Glinos K, et al. Recurrent cough and hemoptysis associated with tracheal capillary hemangioma in an adolescent boy: a case report. J Thorac Cardiovasc Surg, 2007, 134(5): 1366-1367.
24. Zhu L, Wang YQ, Li HC. Extirpation of bronchial hemangioma using bronchoscope: case report. Chin Med J, 2006, 119(3): 259-266.
25. Cordos I, Ulmeanu R, Mihaltan FD, et al. Sequential approach in a case of tracheal hemangioma: segmental tracheal resection after Nd-YAG laser application. Pneumologia, 2005, 54(4): 191-194.
26. Madhumita K, Sreekumar KP, Malini H, et al. Tracheal haemangioma: case report. J Laryngol Otol, 2004, 118(8): 655-658.
27. Irani S, Brack T, Pfaltz M, et al. Tracheal lobular capillary hemangioma: a rare cause of recurrent hemoptysis. Chest, 2003, 123(6): 2148-2149.
28. Zambudio AR, Calvo MJ, Lanzas JT, et al. Massive hemoptysis caused by tracheal hemangioma treated with interventional radiology. Ann Thorac Surg, 2003, 75(4): 1302-1304.
29. 余雪濤, 方偉強. 氣管內海綿狀血管瘤 1 例. 中國實用內科雜志, 2003, 23(5): 310.
30. 張遜, 姚計方, 張澤峰. 氣管血管瘤一例報告. 中國肺癌雜志, 2001, 4(2): 143.
31. So SC, Kwack KK, Park HK, et al. A case of tracheal hemangioma manifested massive hemoptysis. Tuberc Respir Dis, 1999, 47(5): 704-708.
32. Strausz J, Soltesz I. bronchial capillary hemangioma in adults. Pathol Oncol Res, 1999, 5(3): 233-234.
33. Brown JS, Rebeiz EE. Pathologic quiz case 1. Capillary hemangioma. Arch Otolaryngol Head Neck Surg, 1993, 119(6): 700-702.
34. Wigton RB, Rohatgi PK. Isolated bronchial capillary hemangioma: a rare benign cause of hemoptysis. South Med J, 1979, 72(10): 1339-1340.
35. Harding JR, Williams J, Seal RM. Pedunculated capillary hemangioma of the bronchus. Br J Dis Chest, 1978, 72(4): 336-342.
36. 馬蕓, 馬利君, 趙麗敏. 氣道毛細血管瘤病例報告及文獻復習. 中國內鏡雜志, 2014, 20(10): 1116-1118.
37. Sweetser TH. Hemangioma of the larynx. Laryngoscope, 2009, 31(10): 797-806.
38. Ferretti GR, Bithigofer C, Righini CA, et al. Imaging of tumors of the trachea and central bronchi. Radiol Clin North Am, 2009, 47(2): 227-241.
39. 林一丹, 蔣光亮. 胸部血管瘤的臨床特點與外科治療. 中國胸心血管外科臨床雜志, 2013, 20(1): 67-69.
40. 于濤. 血管瘤發病機制的研究進展. 國際口腔醫學雜志, 2007, 34(2): 122-124.
41. Yoshino N, Okada D, Ujiie H, et al. Venous hemangioma of the posterior mediastinum. Ann Thorac Cardiovasc Surg, 2012, 18(3): 247-250.
42. Schneider T, Storz K, Dienemann H, et al. Management of iatrogenic tracheobronchial injuries: a retrospective analysis of 29 cases. Ann Thorac Surg, 2007, 83(6): 1960-1964.
43. Vivas-Colmenares GV, Fernandez-Pineda I, Lopez-Gutierrez JC, et al. Analysis of the therapeutic evolution in the management of airway infantile hemangioma. World J Clin Pediatr, 2016, 5(1): 95-101.
44. Sakr L, Dutau H. Massive hemoptysis: an update on the role of bronchoscopy in diagnosis and management. Respiration, 2010, 80(1): 38-58.

Chinese Journal of Respiratory and Critical Care Medicine

Airway hemangioma: a case report and literature review

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