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+A 57-year-old male was admitted to our hospital with 5 months’ history of massive rectal bleeding (rectorrhagia).
+He had past history of passage of fresh blood mixed stool since last 55 years.
+He first complained of rectal bleeding at the age of 2.
+He went hospital numerous times for this symptom; multiple interventions were done but disease was not cured completely.
+During one of his hospital visit in the past (patient forgot the date), he was diagnosed and treated as hemorrhoids.
+The symptom was relieved for a while and then recurred again.
+Thirty years ago, he was diagnosed as rectal hemangioma and managed with cryotherapy in our hospital.
+That could also just relieve the symptom for few years and it recurred again.
+Then, he consulted many other hospitals, but could only get short-term symptomatic relief without proper treatment of the cause.
+Since 5 months, the severity of rectal bleeding increased.
+He then went to local hospital where intervention was done to control bleeding and symptomatic treatment was done with intravenous fluid and blood transfusion.
+He finally referred to our hospital for further evaluation.
+Intermediate rectal bleeding of fresh blood was presented on admission.
+Patient complained of dizziness on standing, shortness of breath, and palpitation on walking a short distance.
+There was no history of similar illness in family.
+On general examination, patient appeared pale, but his heart rate and blood pressure were within normal limit during rest.
+He lost 5 kg of his body weight during last 5 months.
+On rectal examination, fresh blood was seen around anal region and soft mass was felt on digital rectal examination.
+On proctoscopy, anal cavity and rectum were seen filled with fresh blood, but active site of bleeding, polyp, or ulcer was not detected.
+On laboratory examination, red blood cell count was 3.09 × 1012/L (Normal: 4.32–5.72 × 1012/L) and hemoglobin was 86 g/L (Normal: 135–175 g/L).
+All other parameters were within normal limit.
+None enhanced CT showed isodense (35 HU) homogenous bowel wall thickening that on contrast-enhanced CT venous phase enhances heterogeneously.
+Multiple calcifications were seen in the thickened bowel wall and around the peri-rectal area.
+Lesion was seen extending from distal sigmoid to whole of the rectum (Fig.1).
+Multiple hypodense lesions were also seen in spleen (Fig.2).
+After initial management of anemia, the patient underwent abdominal laparotomy followed by surgical excision.
+During surgery, 25 cm long lesion was found extending from distal sigmoid to whole of the rectum.
+Whole of the rectum and part of the sigmoid colon were excised and sigmoid-anus anastomosis was done.
+Postsurgical histopathological examination of excised specimen showed submucosal multiple thin-walled vessel of varying size with interposed stroma.
+Some vessels lumen consisted of blood cells (consistent with blood vessel), whereas other consisted of clear fluid (consistent with lymph vessel).
+Immunohistochemistry of specimen showed endothelial cells positive for CD 31 and CD 34.
+Some cells were positive for D2–40, while others were negative for D2–40 (Fig.3).
+On the basis of histopathological report and immunohistochemistry, hemolymphangioma was diagnosed.
+The surgery, which followed by complication (intestinal infection), was well managed and the patient was discharged from hospital on the 23rd day of surgery.
+Then after, no further complication or recurrence was noticed during 6 months’ follow-up.
+This study was approved by the First Affiliated Hospital of Sun Yat-Sen University Institutional Review Board.
+Written consent for this case report was obtained from the patient.