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Abstract
Solitary rectal ulcer syndrome (SRUS) is a rare disorder associated with abnormal defaecation. It typically presents with rectal bleeding, mucorrhoea, tenesmus and an altered bowel habit, often mirroring symptoms of ulcerative proctitis. SRUS can be identified on endoscopy by macroscopic findings of: solitary or multiple ulcerated or polypoid lesions 4–10 cm from the anal margin. Mucosal prolapse syndrome has recently been proposed as a more accurate diagnostic description. Adult incidence is 1 per 100,000 with the condition thought to be rarer in children. No guidelines exist on managing SRUS.
A 15-year-old boy with a background of autism, anxiety and ADHD was referred to our rapid access clinic with a 7-month history of rectal bleeding and mucous discharge. His main symptoms were altered bowel habit, weight loss and on some days extreme faecal urgency and frequency, resulting in toileting 40 times in a 24-hour period. His inflammatory markers, albumin and haemoglobin were within normal range and his faecal calprotectin was <20ug/g. His MRI enterogram showed rectal wall thickening suggestive of proctitis. An urgent ileocolonoscopy was performed with macroscopic findings (figure 1) and histology confirming SRUS. The histology showed focal superficial necrosis, patchy mild fibrosis with moderately dilated crypts, thickening of muscularis mucosa with extension of smooth muscle fibres into lamina propria.
He was commenced on prednisolone suppositories and subsequently foam enemas which did not alleviate his symptoms. Over the next few months, he lost 10% of his bodyweight despite regular community dietetic review and increased nutritional supplements. Inpatient admission was required to establish nasogastric tube feeding which he currently continues.
His ileocolonoscopy was repeated after 3 months due to limited symptomatic improvement, alongside an OesophagoGastroDuodenoscopy (OGD). His SRUS remained unchanged macroscopically and histologically. Oesophageal histology showed eosinophilic abscesses, 23 eosinophils per high powered field and basal cell hyperplasia. He was commenced on orodispersible budesonide to treat unrecognised Eosinophilic Oesophagitis (EoE). Our gastroenterology psychologist trialled defaecation behavioural therapy which had limited success due to the patients existing autism and ADHD.
Despite his EoE symptoms improving his lower gastrointestinal symptoms have worsened and he is now experiencing faecal stress incontinence. He no longer leaves the house and the family’s quality of life is poor. He has been referred to adult surgical colleagues to consider surgical approaches including laparoscopic rectopexy, mucosal resection and stoma formation.
In summary, first line approaches for SRUS include treating any existing constipation and modifying defaecation behaviour. We present a particularly challenging case not amenable to medical or psychological behaviour modifications. Dramatic weight loss and an association with EoE has not been previously described. The prognosis for this case remains unclear and we thank the patient and their family for allowing us to raise awareness, to help inform clinicians and families dealing with this rare and disabling condition.
Endoscopic views of the Rectum)