Published online Jan 6, 2020. doi: 10.12998/wjcc.v8.i1.157
Peer-review started: October 4, 2019
First decision: October 23, 2019
Revised: October 31, 2019
Accepted: November 15, 2019
Article in press: November 15, 2019
Published online: January 6, 2020
Processing time: 94 Days and 15 Hours
Isolated gastrointestinal venous malformations (GIVMs) are extremely rare congenital developmental abnormalities of the venous vasculature. Because of their asymptomatic nature, the diagnosis is often quite challenging. However, as symptomatic GIVMs have nonspecific clinical manifestations, misdiagnosis is very common. Here, we report a case of isolated diffuse GIVMs inducing mechanical intestinal obstruction. A literature review was also conducted to summarize clinical features, diagnostic points, treatment selections and differential diagnosis in order that doctors may have a comprehensive understanding of this disease.
A 50-year-old man presented with recurrent painless gastrointestinal bleeding for two months and failure to pass flatus and defecate with nausea and vomiting for ten days. Digital rectal examination found bright red blood and soft nodular masses 3 cm above the anal verge. Computed tomography showed that part of the descending colon and rectosigmoid colon was thickened with phleboliths in the intestinal wall. Colonoscopy exhibited bluish and reddish multinodular submucosal masses and flat submucosal serpentine vessels. Endoscopic ultrasonography showed anechoic cystic spaces within intestinal wall. The lesions were initially thought to be isolated VMs involving part of the descending colon and rectosigmoid colon. Laparoscopic subtotal proctocolectomy, pull-through transection and coloanal anastomosis and ileostomy were performed. Histopathology revealed intact mucosa and dilated, thin-walled blood vessels in the submucosa, muscularis, and serosa involving the entire colorectum. The patient recovered with complete symptomatic relief during the 52-mo follow-up period.
The diagnosis of isolated GIVMs is challenging. The information presented here is significant for the diagnosis and management of symptoms.
Core tip: Few cases of gastrointestinal venous malformations (GIVMs) responsible for intestinal obstruction in adults have been described. We present a rare case of isolated GIVMs involving the colorectum in a patient with symptoms of intestinal obstruction. Digital rectal examination, enhanced computed tomography, colonoscopy, endoscopic ultrasonography, barium contrast examination and selective mesenteric angiography contributed to a precise diagnosis. Laparoscopic subtotal proctocolectomy, pull-through transection and coloanal anastomosis were performed. The patient did not develop any serious complications during the follow-up period. We have also summarized the information obtained from 152 cases reported to date, which is important in assisting physicians with the diagnosis and treatment.