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CASE REPORT Table of Contents   
Year : 2009  |  Volume : 52  |  Issue : 3  |  Page : 392-394
Sclerosing hemangioma with lymph nodal metastases


Department of Pathology (Cardiovascular & Thoracic Division), Seth G. S. Medical College, Mumbai, India

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Date of Web Publication12-Aug-2009
 

   Abstract 

A case of sclerosing hemangioma of the lung is reported in a young male, who presented with recurrent cough and streaky hemoptysis for three years. The tumor was situated in the right upper lobe and was large (over 9 cm), multicentric and associated with metastases to the regional lymph nodes. To the best of our knowledge, only 14 cases with such metastases have been reported.

Keywords: Lung, lymph node metastases, sclerosing hemangioma

How to cite this article:
Vaideeswar P. Sclerosing hemangioma with lymph nodal metastases. Indian J Pathol Microbiol 2009;52:392-4

How to cite this URL:
Vaideeswar P. Sclerosing hemangioma with lymph nodal metastases. Indian J Pathol Microbiol [serial online] 2009 [cited 2019 Oct 18];52:392-4. Available from: http://www.ijpmonline.org/text.asp?2009/52/3/392/55004



   Introduction Top


Sclerosing hemangioma (SH) is a rare tumor that has been categorized under the miscellaneous group of pulmonary neoplasms in the recent World Health Organization (WHO) classification. [1] It is a misleading designation for a tumor that originates from the pulmonary epithelial cells. The lesion usually presents as a sub-pleural, slow-growing 'coin lesion' in the lower lobes of middle-aged females with a benign course. We report a multi-focal SH in a young male with lymph node metastases.


   Case Report Top


A 23-year-old male had reported cough with minimal mucoid expectoration for many years. This, for the past four months, was associated with streaky hemoptysis, occasional episodes of dyspnea at night, and loss of weight and appetite. Anti-tuberculous therapy had been instituted thrice (1986, 1997 and 2002) in the past for similar complaints with no clinical improvement. General and systemic examinations were unremarkable. Hematological and biochemical parameters were normal. Chest radiography had revealed a right upper zone, multiple conglomerated mass lesion with irregular calcification [Figure 1]a. Computed tomographic (CT) scan showed an enhancing mass in the posterior segment of the right upper lobe with multiple acinar foci in the periphery [Figure 1]b, splaying and compression of the bronchi and calcified lymph nodes. Bronchoscopic cytology was normal. A CT-guided mass biopsy revealed strips of tissue composed of alveolar septa lined by plump cuboidal to columnar epithelial cells. At places, there were papillae or stromal sclerosis. The impression was a bronchiolo-alveolar tumor.

A right upper lobectomy was performed. The lobe was expanded and measured 12x 9x 7 cm. The pleura was moderately thickened and opacified with multiple yellowish protrusions, especially in the apical segment. The consistency was firm. The cut-surface showed a large (9x 7x 5 cm), well-circumscribed, homogeneous ivory white tumor almost occupying the entire lobe [Figure 1]c. The tumor was lobulated and bulged above the cut-surface; the lobules were separated by either fibrous septa or compressed parenchyma. Interestingly, the surrounding lung had multiple, small, white, firm, satellite nodules. On further serial slices, there were three large nodules that appeared distinct from the larger tumor mass. One of them was hemorrhagic, while the two others were pearly white.

Histologically [Figure 2]a-c, the tumor was composed of two cell types in several patterns. The prominent pattern was papillary, with papillae lined by flattened to cuboidal epithelium that focally continued with the bronchiolar epithelium. Large polygonal cells with moderate light pink cytoplasm and vesicular nuclei were also disposed in sheets, interspersed by splotchy foci of calcification and collections of foamy macrophages. This constituted the solid pattern. The macrophages also contained finely dispersed yellow brown hemosiderin pigment. The hemangiomatous pattern was characterized by dilated blood-filled spaces, amidst sheets of polygonal cells seen in sections from the hemorrhagic nodule. The two other nodules comprised of large sclerosis with epithelial-lined clefts, simulating ancient fibroadenoma (the sclerotic pattern). Tumor was also seen to abut the larger airways.

Hilar lymph nodes were calcified. Multiple, small nodular areas of sclerosis with epithelial-lined clefts and occasional papillae [Figure 2]d were identified in the sub-capsular region. A diagnosis of SH with lymph node metastases was offered. The patient had an uneventful postoperative course and was discharged on the 12 th day.


   Discussion Top


SH was a term coined by Liebow and Hubbell [2] to describe a rare benign tumor of uncertain histogenesis. Though several studies have advocated varying cells of origin and characteristics, the pulmonary cell of origin has been proved beyond doubt by immunohistochemical demonstration of thyroid transcription Factor 1 (TTF 1). [3] Since old habits die hard, the original christening continues to be preferred over "sclerosing pneumocytoma" in the WHO classification.

These tumors have a propensity to occur in adult women (mean age 46.2 years). Since they are frequently asymptomatic, they are detected on chest radiography. [4] Sometimes, there may be associated cough with hemoptysis. This patient case had recalcitrant cough for many years and was repeatedly administered anti-tuberculous therapy without much improvement. It is, hence, probable that the symptoms may have been due to the tumor, which explains the large size; there were satellite nodules as well. In a large series of 100 cases, the average size of SH was 3.5 cm ('coin lesion' on X-ray) and only 4 % were multicentric. [4]

The tumor is composed of two cell types: polygonal (also called round or interstitial or lesional) cells and the cuboidal (also called lining or surface cells). [4],[5] These cells are arranged in varying patterns such as in sheets or papillae, hemangiomatous or sclerotic patterns, [4] all of which were seen in the present tumor. Sampling of one such area creates diagnostic difficulty on fine needle aspirations, frozen sections as well as in biopsy interpretations and hence these lesions may be confused with other clear cell or papillary tumors. [4] Though both the cell types are TTF 1-positive, [3] the former are cytokeratin-negative and epithelial membrane antigen-positive while the latter cells are positive for both markers. [5] We have not performed immunohistochemistry in this case.

Interestingly, there were sub-capsular deposits of sclerotic tumor in many hilar lymph nodes. So far, there have been 14 reported in the literature of such sub-capsular metastases detected microscopically. [3],[4] [6],[7],[8],[9],[10],[11],[12],[13],[14] Despite this, the tumor pursues a benign course and therapy is restricted to surgical resection or even thoracoscopic enucleation. Dystrophic calcification was also present. Upper lobe shadow and calcified lymph nodes can simulate tuberculosis on chest X-ray, a common finding in most Indians. In our country when presumptive diagnosis of tuberculosis is made often on the basis of radiology, clinicians must be aware of potentially curable conditions like SH.

 
   References Top

1.Travis WD, Colby TV, Corrin B, Shimosato V, Brambilla E and collaborators from 14 countries. WHO histologic typing of lung and pleural tumors. 3 rd ed. Geneva World Health Organization; 1999.   Back to cited text no. 1    
2. Liebow AA, Hubbell DS. Sclerosing hemangioma (histiocytoma, xanthoma) of the lung. Cancer 1956;9:53-7.  Back to cited text no. 2    
3.Chan AC, Chan JK. Pulmonary sclerosing hemangioma consistently expresses thyroid transcription factor-1 (TTF-1). A new clue to its histogenesis. Am J Surg Pathol 2000;24:1531-6.  Back to cited text no. 3    
4.Devouassoux-Shisheboran M, Hyashi 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.Rodriguez-Soto J, Colby TV, Rouse RV. A critical examination of the immunophenotype of pulmonary sclerosing hemangioma. Am J Surg Pathol 2000;24:442-50.  Back to cited text no. 5    
6.Tanaka I, Inoue M, Matsui Y. A case of pneumocytoma (so-called sclerosing hemangioma) with lymph node metastases. Jpn J Clin Oncol 1986;16:77-86.  Back to cited text no. 6    
7.Miyagawa-Hayashino A, Tazelaar HD, Langel DJ, Colby TV. Pulmonary sclerosing hemangioma with lymph node metastases: a0 report of 4 cases. Arch Pathol LabMed 2003;127:321-5.  Back to cited text no. 7    
8.Kim KH, Sul HJ, Kang DY. Sclerosing hemangioma with lymph metastasis. Yonsei Med J 2003;44:150-4.  Back to cited text no. 8    
9.Yano M, Yamakawa Y, Kinyama M, Hara M, Murase T. Sclerosing hemangioma with metastases to multiple nodal stations. Ann Thorac Surg 2002;73:781-3.  Back to cited text no. 9    
10.Spencer H, Nambu S. Sclerosing hemangiomas of the lung. Histopathology 1986;10:477-87.  Back to cited text no. 10    
11.Chan NG, Melega DE, Inculet RI, Shepherd JG. Pulmonary sclerosing hemangioma with lymph node metastases. Can Respir J 2003;10:391- 2.  Back to cited text no. 11    
12.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. 12    
13.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. 13    
14.Komatsu T, Fukuse T, Wada H, Sakurai T. Pulmonary sclerosing hemangioma with pulmonary metastasis. Thorac Cardiovasc Surg 2006;54:348-9.  Back to cited text no. 14    

Top
Correspondence Address:
Pradeep Vaideeswar
Department of Pathology (Cardiovascular & Thoracic Division), Seth G. S. Medical College, Parel, Mumbai - 400 012
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.55004

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    Figures

  [Figure 1], [Figure 2]

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