Abstract
BackgroundPulmonary sclerosing hemangioma (SH) is a rare tumor of the lung predominantly affecting Asian women in their fifth decade of life. SH is thought to evolve from primitive respiratory epithelium and mostly shows benign biological behavior; however, cases of lymph node metastases, local recurrence and multiple lesions have been described.Case PresentationWe report the case of a 21-year-old Caucasian male with a history of locally advanced and metastatic rectal carcinoma (UICC IV; pT4, pN1, M1(hep)) that was eventually identified as having hereditary non-polyposis colorectal cancer (HNPCC, Lynch syndrome). After neoadjuvant chemotherapy followed by low anterior resection, adjuvant chemotherapy and metachronous partial hepatectomy, he was admitted for treatment of newly diagnosed bilateral pulmonary metastases. Thoracic computed tomography showed a homogenous, sharply marked nodule in the left lower lobe. We decided in favor of atypical resection followed by systematic lymphadenectomy. Histopathological analysis revealed the diagnosis of SH.ConclusionsCases have been published with familial adenomatous polyposis (FAP) and simultaneous SH. FAP, Gardner syndrome and Li-Fraumeni syndrome, however, had been ruled out in the present case. To the best of our knowledge, this is the first report describing SH associated with Lynch syndrome.
Highlights
Pulmonary sclerosing hemangioma (SH) is a rare tumor of the lung predominantly affecting Asian women in their fifth decade of life
Cases have been published with familial adenomatous polyposis (FAP) and simultaneous SH
To the best of our knowledge, this is the first report describing SH associated with Lynch syndrome
Summary
We present the first case of pulmonary SH in a young Caucasian male and in a patient suffering from Lynch syndrome. It might be speculated that SH did not just incidentally co-occur with the patient’s CRC. From this unlikely concurrence we assume that the underlying Lynch syndrome might have abetted the arising of the patient’s SH and hypothesize a common cause for these rare events. Author details 1Department of Surgery, University of Munich, Campus Grosshadern, Germany. Authors’ contributions TSS, PNK and AK collected all patient’s history data with substantial contribution of WET, MAK, RAH and KWJ. TSS, PNK and AK drafted the manuscript with committed and dedicated review and discussion of WET, MAK, RAH and KWJ. Competing interests The authors declare that they have no competing interests
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