Unusual cause of 55 years of rectal bleeding: hemolymphangioma (a case report)

RATIONALE: Hemolymphangioma is a rare developmental error of combined blood and lymphatic vasculature. To the best of our knowledge, there is only one case of rectal hemolymphangioma reported in Pubmed. Our case probably is the first reported rectal hemolymphangioma with computed tomography (CT) evaluation. PATIENT CONCERNS AND DIAGNOSIS: A 57-year-old male was presented to our hospital with 55 years of long history of episodic rectal bleeding. Past medical history showed numerous hospital visits for similar illness. Multiple diagnoses were made and different treatment modalities were applied for his benefit, but none of them relieved the symptoms permanently. He was then referred to our hospital. On admission, he was presented with intermediate rectal bleeding of fresh blood. CT examination showed isodense homogenous rectal wall thickening with heterogeneous enhancement on contrast examination. Multiple calcifications were seen in and around the lesion. INTERVENTIONS AND OUTCOMES: He underwent open abdominal surgery with total surgical excision of the lesion. Post-surgical histopathological examination of excised specimen showed submucosal multiple thin-walled vessel of varying size, some consistent with blood vessel and other with lymph vessel, thus diagnosis of hemolymphangioma was made. Follow-up for 6 months showed no recurrence. LESSIONS: Hemolymphangioma is a benign developmental lesion. Radiological findings can be challenging and range from benign cystic lesion to aggressive lesion mimicking malignancy. Therefore, combined clinical history, radiological findings, and continuous follow-up can help make proper diagnosis and provide prompt and accurate treatment.