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Year : 2013  |  Volume : 9  |  Issue : 4  |  Page : 748-750

Diagnosis of sclerosing hemangioma of lung: Don't rely on fine-needle aspiration cytology diagnosis alone

1 Department of Pulmonary Medicine, Burdwan Medical College, Burdwan, India
2 Department of Pulmonary Medicine, NRS Medical College and Hospital, 138 AJC Bose Road, Shealdah, Kolkata - 700 014, West Bengal, India

Date of Web Publication11-Feb-2014

Correspondence Address:
Kaushik Saha
Rabindra Pally, 1st Lane, P.O. - Nimta, Kolkata - 700 049, West Bengal
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/0973-1482.126486

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 > Abstract 

Sclerosing hemangioma is a rare variety of benign pulmonary neoplasm. It usually presents as asymptomatic, solitary, peripheral, circumscribed lesions in middle-aged women. Here, we describe a 46-year-old woman presenting to us for evaluation of right parahilar lung mass. Previous chest radiography done 10 years back showed a lung mass of almost similar size. Computed Tomography (CT)-guided fine-needle aspiration cytology (FNAC) was suggestive of adenocarcinoma of lung. A well-circumscribed, capsulated, ovoid mass measuring 5.6 cm × 4 cm × 3 cm, adjacent to the transverse fissure of the right lung was excised by lateral thoracotomy. Histopathological examination along with immunohistochemistry was suggestive of sclerosing hemangioma of lung. A pathologist must consider the clinicoradiological features before coming to a final diagnosis of lung malignancy from FNAC. Whenever there is any confusion regarding lung mass, thoracotomy must be done for arriving at an exact diagnosis from histopathology.

Keywords: Lung, sclerosing hemangioma, woman

How to cite this article:
Saha K, Sit NK, Jash D, Maji A. Diagnosis of sclerosing hemangioma of lung: Don't rely on fine-needle aspiration cytology diagnosis alone. J Can Res Ther 2013;9:748-50

How to cite this URL:
Saha K, Sit NK, Jash D, Maji A. Diagnosis of sclerosing hemangioma of lung: Don't rely on fine-needle aspiration cytology diagnosis alone. J Can Res Ther [serial online] 2013 [cited 2021 Sep 24];9:748-50. Available from: https://www.cancerjournal.net/text.asp?2013/9/4/748/126486

 > Introduction Top

Sclerosing hemangioma is a rare benign neoplasm of lung with uncertain histogenesis, first described by Liebow and Hubbel in 1956. [1] In WHO classification, it has been classified under miscellaneous group of pulmonary neoplasms. [2] It commonly occurs in middle-aged women, as asymptomatic solitary well-circumscribed lesion located in the periphery of the lung. [3] Histologically, the characteristics have been well described as papillary, solid, sclerotic, or hemorrhagic patterns. Malignant lung tumor is a close differential diagnosis of sclerosing hemangioma. Here, we report a case of sclerosing hemangioma of lung in a 46-year-old woman initially diagnosed as adenocarcinoma of lung from fine-needle aspiration cytology (FNAC).

 > Case Report Top

A 46-year-old woman was admitted with a history of chest pain for 3 months. Physical examination and routine blood investigation at the time of admission revealed no positive findings. Chest X-ray (CXR) showed a well-circumscribed mass in right lung in the region of middle lobe close to hilum. Contrast-enhanced computed tomography (CECT) thorax showed a solitary well-defined heterogeneously enhancing soft tissue density lesion in the right middle lobe beside the heart measuring about 5.3cm × 5.1 cm × 4.8 cm in size [Figure 1]a.
Figure 1: Contrast-enhanced computed tomography thorax (a) showing a well-defined heterogeneously enhancing soft tissue density lesion in the right middle lobe measuring about 5.3 cm × 5.1 cm × 4.8 cm and contrast-enhanced computed tomography thorax of 10 years back (b) showing a well-bordered lung mass measuring 4.47 cm × 4.48 cm in the right middle lobe

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She attended a local chest clinic 10 years back due to right-sided chest pain. Review of her previous medical records revealed presence of a lung mass in a CXR dated 10 years back. CECT thorax at that time showed a solitary lung mass measuring 4.47 × 4.48 cm, fairly enhancing 49 hounsfield unit (HU) in noncontrast, and 87 HU in post-contrast values in the same position like the present CECT thorax [Figure 1]b. She received oral antibiotics and analgesic for her symptoms at that time and achieved symptomatic relief. CT-guided FNAC of that mass revealed a non-specific inflammation. She remained asymptomatic for last 10 years and no follow-up visits were done.

CT-guided FNAC of the lung mass after presentation to us showed cellular smear in multiple clumps and dense atypical bronchial epithelial cells on the inflammatory background [Figure 2]. The cells had mildly pleomorphic prominent hyperchromatic nuclei. Occasional clumps of cells showed vacuolated cytoplasm. The overall cytomorphological features were suggestive of well-differentiated adenocarcinoma. However, the long duration along with little increase in size of the mass in last 10 years raised the suspicion of benign neoplasm. Fiber-optic bronchoscopy was within normal limits. Thoracotomy was done for complete excision of the mass to identify the exact etiology. A well-circumscribed capsulated ovoid mass measuring 5.6 cm × 4 cm × 3 cm adjacent to the transverse fissure of the right lung was excised by lateral thoracotomy [Figure 3]. The cut surface showed brownish and hemorrhagic areas. The section showed a lesion composed of blood-filled spaces lined by epitheloid cells that were surrounded by round, oval, or spindle-shaped cells admixed with a fair number of foamy histiocytes with absence of mitotic figures [Figure 4]. The features were compatible with sclerosing hemangioma of lung. Upon performing immunohistochemistry, the cells showed a typical expression of thyroid transcription factor (TTF) 1 and epithelial membrane antigen and were negative for cytokeratin markers. Cells did not express CD34, but revealed a high vessel density. The diagnosis of sclerosing hemangioma was established.
Figure 2: Computed tomography-guided fine-needle aspiration cytology of the lung mass showing cellular smear in multiple clumps and dense atypical bronchial epithelial cells on the inflammatory background with occasional clumps of cells having vacuolated cytoplasm and mildly pleomorphic prominent hyperchromatic nuclei suggestive of well-differentiated adenocarcinoma (H and E stain, ×100)

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Figure 3: A well-circumscribed capsulated ovoid mass 5.6 cm × 4 cm × 3 cm excised by thoracotomy

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Figure 4: Excisional biopsy of lung mass showing blood-filled spaces lined by epitheloid cells that are surrounded by round, oval, or spindle-shaped cells admixed with a fair number of foamy histiocytes suggestive of sclerosing hemangioma of lung (H and E stain, ×100)

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 > Discussion Top

Since the original description by Liebow and Hubbell, multiple theories have been proposed for its histogenesis including mesothelial, mesenchymal, epithelial, and neuroendocrine origin. Current studies strongly suggest origin of sclerosing hemangioma from primitive respiratory epithelium. [4] A variety of terms such as pneumocytoma, sclerosing pneumocytoma, and papillary pneumocytoma have been proposed for this lesion. Although the term sclerosing hemangioma is a misnomer, it has retained its place due to historical reasons. [2] It has a propensity to occur in middle-aged women with female to male ratio of 5:1. [4] Majority of the patients are asymptomatic with the tumor being recognized on screening chest radiographs. Sometimes, the patient may present with hemoptysis, cough, or chest pain. CXR characteristics are sharply marked, well defined, solitary, peripheral, homogenous mass without any lobar predilection. [5] On CECT, there is contrast-enhancement of the mass with little or no peripheral calcification. There is absence of infiltration of surrounding tissues along with absence of cavities. [6] On gross examination, sclerosing hemangioma presents as well-circumscribed, yellow-tan peripheral mass with occasional areas of hemorrhage. [4] Histologically, this tumor is highly vascular, partially hemorrhagic, papillary, folding with cuboidal superficial proliferating epithelium in between the collagen fibrils and round cells. They consist of two epithelial cell types, surface cells and round cells, and four architectural patterns, papillary, sclerotic, solid, and hemorrhagic. 95% of sclerosing hemangioma possess three architectural patterns, most commonly papillary, sclerotic, and solid, and all contain at least 2 patterns. [4] In the papillary pattern, complex papillae are composed of surface cells covering a stroma of round cells. The sclerotic pattern is characterized by hyalinized collagen in papillae, in solid areas, or at the periphery of hemorrhagic areas. Sheets of round cells bordered by surface cells form the solid pattern. Finally, large blood-filled spaces comprise the hemorrhagic pattern. [4],[7] Several immunohistochemical studies established that both the surface cells and the round cells are characterized by nuclear expression of TTF-1 and membranous or cytoplasmic expression of EMA in nearly all cases. [4],[8] The histological differential diagnosis included benign and malignant lung tumors and metastatic carcinoma. The distinction between benign and malignant may be difficult particularly at the time of FNAC. Architectural variation and two epithelial cell types may not be readily appreciated leading to misdiagnosis of malignancy. In some cases, adenocarcinoma like cells may be found within the mass to add more confusion. [9] Sclerosing hemangioma of lung is generally considered benign and surgical excision is curative without requirement of additional treatment. Presence of lymph-node metastasis does not affect treatment and prognosis. [10]

Sclerosing hemangioma should be in mind of a clinician as differential diagnosis in patients having lung mass for long duration. Benign clinical course over the years and typical radiologic and macroscopic features help in suspecting sclerosing hemangioma. The pathologist must be aware of this before misdiagnosing it as malignancy. FNAC alone cannot be accepted as the final definitive diagnosis for any lung tumor. Clinical judgment is still the most important element in managing our patients. If we rely on FNAC and imaging finding alone, patients who are medically not fit for surgery could have received some unnecessary nonsurgical treatment (e.g., radiotherapy, chemotherapy, targeted therapy, etc.). The clinician should also be careful enough in explaining the situation to patients and their relatives to avoid psychological trauma to the patient. In conclusion, whenever the patients with lung mass become symptomatic or whenever there is any confusion regarding the diagnosis, exploratory thoracotomy should always be done as it allows exact diagnosis and treatment for benign lesions.

 > References Top

1.Liebow AA, Hubbell DS. Sclerosing hemangioma (histiocytoma, xanthoma) of the lung. Cancer 1956;9:53-75.  Back to cited text no. 1
2.Devouassoux-Shisheboran M, Nicholson GA, Leslie K, Niho S. Sclerosing hemangioma. In: Travis WD, Brambilla E, Muller-Hermelink HK, Harris CC, editors. Pathology and genetics: tumors of the lung, pleura, thymus and heart. Lyon, France: IARC; 2004. p. 115-7.   Back to cited text no. 2
3.Iyoda A, Hiroshima K, Shiba M, Haga Y, Moriya Y, Sekine Y, et al. Clinicopathological analysis of pulmonary sclerosing hemangioma. Ann Thorac Surg 2004;78:1928-31.  Back to cited text no. 3
4.Devouassoux-Shisheboran M, Hayashi T, Linnoila RI, Koss MN, Travis WD. A clinicopathologic study of 100 cases of pulmonary sclerosing hemangioma with immunohistochemical studies: TTF-1 is expressed in both round and surface cells, suggesting an origin from primitive respiratory epithelium. Am J Surg Pathol 2000;24:906-16.  Back to cited text no. 4
5.Chung MJ, Lee KS, Han J, Sung YM, Chong S, Kwon OJ. Pulmonary sclerosing hemangioma presenting as solitary pulmonary nodule: Dynamic CT findings and histopathologic comparisons. AJR Am J Roentgenol 2006;187:430-7.  Back to cited text no. 5
6.Cheung YC, Ng SH, Chang JW, Tan CF, Huang SF, Yu CT. Histopathological and CT features of pulmonary sclerosing haemangiomas. Clin Radiol 2003;58:630-5.  Back to cited text no. 6
7.Gal AA, Nassar VH, Miller JI. Cytopathologic diagnosis of pulmonary sclerosing hemangioma. Diagn Cytopathol 2002;26:163-6.  Back to cited text no. 7
8.Illei PB, Rosai J, Klimstra DS. Expression of thyroid transcription factor-1 and other markers in sclerosing hemangioma of the lung. Arch Pathol Lab Med 2001;125:1335-9.  Back to cited text no. 8
9. Kim GY, Kim J, Choi YS, Kim HJ, Ahn G, Han J. Sixteen cases of sclerosing hemangioma of the lung including unusual presentations. J Korean Med Sci 2004;19:352-8.  Back to cited text no. 9
10. Katakura H, Sato M, Tanaka F, Sakai H, Bando T, Hasegawa S, et al. Pulmonary sclerosing hemangioma with metastasis to the mediastinal lymph node. Ann Thorac Surg 2005;80:2351-3.  Back to cited text no. 10


  [Figure 1], [Figure 2], [Figure 3], [Figure 4]


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