Solitary Rectal Ulcer Syndrome in Children: A Case Series Study

Abstract

Information on solitary rectal ulcer syndrome (SRUS) in children is limited and based on case reports only. This study was undertaken with the objective of describing the clinical history, symptoms, diagnostic work-up, and treatment of a large case series of pediatric patients with SRUS. The study was multi-center and retrospective. All pediatric endoscopists in Poland were invited to participate in the study and were asked to look through their endoscopic databases to identify SRUS cases from the last 10 years. The charts of SRUS patients were reviewed with respect to demographic data, and endoscopic and histological findings. Additionally, treatment methods and outcomes were assessed. In total, 31 patients (18 males, mean age of 13 years, range 5-18 years) were included in the study. All patients reported rectal bleeding. Other common symptoms included: abdominal pain (64.5%), perianal pain (54.8%), and passage of mucus (51.6%). The diagnostic work-up lasted from 1 to 48 months. Colonoscopic findings revealed rectal ulceration in 96.8% of patients. Therapeutic approaches included: high fiber diet (64.5%), laxatives (54.8%), topical corticosteroids (63.3%), 5-aminosalicylates administered orally and topically (29.1% and 96.8%, respectively), sucralfate (9.7%), and a biofeedback training (6.6%). Endoscopic argon plasma coagulation was performed in 2 patients and surgical intervention was necessary in 4 of them. Treatment was unsuccessful in 36% of patients. The findings of this study indicate that SRUS is rare in pediatric population, its diagnosis may be considerably delayed, and the treatment applied is often ineffective.

Solitary Rectal Ulcer Syndrome in Children

“…Clinical presentations of our study are almost similar to the previous studies by Kowalsk-Duplaga et al (15) of 31 children and Suresh et al ( 13) of 22 children. The main differences are the history of excessive straining during defecation which we were able to elicit in almost all cases and attempted manual evacuation of stool which was present in 50% of our cases while the figures were 45% to 64% and 19% to 27%, respectively, in previous studies (15,13). Recently, Dehghani et al (16) reported similar clinical features and colonoscopic findings in a study of 55 children.…”

“…Blackburn et al (18) showed a good response to behavioral modification alone in 7 of 8 children with SRUS. Kowalsk-Duplaga et al (15) used topical steroids in 19 of 31 children and showed healing (full or partial) in 14 (63.2%) cases. Dehghani et al (12) showed complete relief of symptoms in 7 (58.3%) of 12 children treated with sucralfate enema.…”

“…First time it was described in 1829 (7); however, proper clinical and histological characterization was made in 1969 only (2). The estimated incidence of SRUS in adults ( 8) is 1 in 100,000 but pediatric publications comprised mainly of case reports and case series (9)(10)(11)(12)(13)(14)(15). The largest pediatric series of SRUS so far is of 55 cases from Iran (16).…”

A Clinical Entity Often Missed—Solitary Rectal Ulcer Syndrome in Children

“…Intermittent rectal bleeding and/or mucorrhea were the commonest presentation similar to other reported series (7,(11)(12)(13)(14)(15) Predisposing factors for SRUS like constipation (hard stool), straining during defecation were present in majority of case series (7,12,(14)(15)(16). The other presenting symptom which signifies rectal diseases like tenesmus, rectal prolapse were present in varying proportions (7,12,(14)(15)(16)(17).…”

Challenges in Differentiating Between Solitary Rectal Ulcer Syndrome and Inflammatory Bowel Disease in the Pediatric Population

“…If neither a conservative method nor biofeedback is therapeutic, and if patients have a rectal prolapse or intussusception, surgical intervention may be considered such as ulcer excision or treatment of the rectal prolapse or rectal intus-susception [13]. If patients do not have a rectal prolapse or intussusception, but neither a conservative method nor biofeedback is therapeutic, local excision may be considered for symptomatic relief [15,16]. Both SRUS cases followed a conservative treatment plan where the former was treated with mesalamine, docusate, and increasing fiber intake.…”