A rare case of pulmonary sclerosing hemagioma with lymph node metastasis and review of the literature

Abstract

Pulmonary sclerosing hemagioma (SH) is an uncommon tumor with malignance potential. Clinically this disease is regarded as benign but extremely rare cases can have lymph node metastasis. Up to date, there have been only very few reports concerning SH with lymph node metastasis. In this paper we reported one pulmonary SH case with lymph node metastasis and additionally overviewed the clinical and pathological features of SH. A young-aged female was found incidentally to have a nodule in the right upper lung. This patient presented no cough, no hemoptysis and chest pain. Computed tomography (CT) scan indicated a large mass in the right upper lung and enlarged lymph nodes in the right hilum. The patient underwent lobectomy of the right upper lung. Histologically, the tumor demonstrated typical features of SH and was consisted of angiomatoid areas, sclerosis, papillary structures lined with cuboidal cells and sheets of round to polygonal cells. Polygonal cells in some solid areas presented abnormal enlarged nuclei and increased karyoplasmic ratio; tumor giant cells were noted; whereas mitosis was not observed. One peribronchial lymph node was noted for SH metastasis and the metastatic tissue were consisted of polygonal cells. Immunohistochemistry (IHC) revealed that both surface-lining cuboidal and polygonal cells expressed EMA and thyroid transcription factor 1 (TTF-1), but were negative for CD34, VIII factor, CD68 and Claratinin. The polygonal cells showed relatively higher expression of Ki-67 and p53 than the surface-lining cells. Postoperatively, the patient received no chemotherapy or radiotherapy and no recurrence 2 years after surgery was noted.

Keywords: Sclerosing hemangioma (SH); lymph node metastasis; pulmonary SH.

Figure 1

Histological findings. A: The polygonal cells were similar in morphology with light-stained eosinophilic cytoplasma (hematoxylin and eosin, × 200). B: The polygonal cells displayed strike pleomorphism, enlarged nuclei and increased karyoplasmic ratio. But mitosis was not observed (hematoxylin and eosin, × 400). C: Metastatic deposits in the lymph node were consisted of polygonal cells (hematoxylin and eosin, × 100).

Figure 2

Immunophenotypic findings. A: The metastatic tumor cells were positive for TTF-1 (immunohistochemistry, × 100). B: The polygonal cells showed higher Ki-67 expression than surface-lining cells (immunohistochemistry, × 400).