Abstract

Pulmonary sclerosing hemangioma (PSH) is a rare benign tumor of the lungs. These tumors are composed of cuboidal surface cells and polygonal stromal cells and show four histological manifestations: hemorrhagic, papillary, solid, and sclerotic. PSH predominantly affects asymptomatic middle-aged women. The tumor often occurs at the intralobar site, and less commonly in the bronchus and mediastinum. PSH is easy to be misdiagnosed preoperatively. In this study, we present in detail the treatment procedures followed for two atypical cases of PSH. Case 1 was a 62-year-old woman bearing a tumor for 15 years. The tumor lesion was found to be located in the oblique fissure of the left lung. PSH was confirmed by surgical resection and postoperative pathological diagnosis. There was no sign of recurrence and metastasis 1.5 years after surgery. Case 2 was a 54-year-old woman diagnosed with bilateral multiple nodules by physical examination. This patient was diagnosed with definite PSH through computed tomography-guided percutaneous lung biopsy. Surgical resection was not performed. The patient also showed no sign of enlarged tumor and metastasis after 2 years of follow-up. Although PSH can be cured by surgical resection, the findings in our cases indicate that surgical resection need not be considered the preferred course of treatment. If PSH is diagnosed before surgery, the patients may survive while bearing the tumor.

Highlights

  • Pulmonary sclerosing hemangioma (PSH) is an uncommon benign tumor, which was first described by Liebow and Hubbell in 1956 [1]

  • PSH can be misdiagnosed as a malignant tumor during intraoperative frozen-section assessment and has a misdiagnosis rate of 25 to 56%, which results in unnecessary extensive surgical procedures and malignant biological behaviors, including local lymph-node

  • Since PSH is known to be composed of cuboidal surface cells and polygonal stromal cells and is derived from type II pneumocytes, we predicted this tumor might have originated in the subpleural lung region of the visceral pleura and spread to the oblique fissure due to prolonged compression and adhesion to the adjacent structure by the tumor

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Summary

Background

Pulmonary sclerosing hemangioma (PSH) is an uncommon benign tumor, which was first described by Liebow and Hubbell in 1956 [1]. Case 1 The patient was an asymptomatic 62-year-old woman diagnosed with a tumor in the left hilum of the lung in 1995, who had refused to undergo treatment (imaging data of the physical examination had been misplaced). The immunohistochemical results were as follows: thyroid nuclear factor 1 (positive; Figure 3B), cytokeratins (negative), vimentin (positive), CD34 (positive), synaptophysin (negative), and epithelial membrane antigen (positive). The results of immunohistochemical analyses were as follows: thyroid nuclear factor 1 (positive), synaptophysin (negative), CD56 (negative), CD68 (positive), cytokeratins (positive), and P63 (negative). This patient was diagnosed with bilateral multiple PSH and was discharged from the hospital without any further adjuvant therapy. Two-year follow-up found no increase in the number and size of the lesions

Discussion
Conclusions
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