An acute hemorrhagic rectal ulcer (AHRU) is a serious medical condition characterized by sudden and painless rectal bleeding resulting from the development of an ulcer in the lower rectum, mainly situated near the dentate line.1 This condition is most commonly observed in bedridden or elderly patients who may have various underlying health complications that contribute to their vulnerability. Due to the presence of comorbidities, patients with AHRUs have a high mortality rate. Specifically, the in-hospital mortality rate has been reported to be as high as 18%. Therefore, the presence of AHRU can be one of the factors affecting poor prognosis.2, 3 Despite its significance, the incidence of AHRU in pediatric populations remains largely unexplored, especially in Japan, where there have been no documented case reports of this condition in children. As such, the prevalence and clinical presentation of AHRU in this demographic remain unknown, creating a gap in clinical knowledge and practice. In this report, we describe the case of AHRU in a child who presented with severe developmental delay and physical disability. This case offered an opportunity to closely observe the progression of this condition over time. A colonoscopy was performed to monitor the chronological changes in ulceration and bleeding. The patient was a 4-year-old boy with drug-resistant epilepsy and severe motor and intellectual disabilities that were present since neonatal life. He underwent gastrostomy at 1 year and 2 months of age and tracheal laryngoplasty at 2 years of age. He was transferred to our hospital after being diagnosed with rectal bleeding while being treated with antibiotics for pneumonia at 3 years of age. A colonoscopy revealed multiple round and irregular ulcers around the entire circumference of the lower rectum just above the dentate line (Figure 1a). Thrombin solution (10,000 units) was sprayed on the base of the ulcers for hemostasis. Additionally, upper gastrointestinal endoscopy and small bowel capsule endoscopy revealed no ulcers in the digestive tract other than the rectum. The histopathological examination of the rectal mucosa revealed mild inflammatory cell infiltration without evidence of fibrotic changes (Figure 1b). The blood test was negative for cytomegalovirus infection. The patient had intractable diarrhea due to the infusion of enteral nutrition and was unable to consume the necessary amount of nutrition. When the child was re-examined by colonoscopy 1 month after the onset of AHRU, the ulcers had not yet healed and recovery was delayed (Figure 1a). The patient's diarrhea improved with the administration of a blended diet instead of an enteral nutrition formula, and his weight and serum albumin levels showed an upward trend (Figure 1c). When the colon was re-examined by colonoscopy 3 months after the onset of AHRU, the ulcer base epithelialized and showed signs of healing (Figure 1a). Additionally, when the patient's colon was re-examined by colonoscopy 5 months after the onset of AHRU, there was no sign of ulcer recurrence (Figure 1a). Most reports of rectal ulcers in children are of solitary rectal ulcer syndrome (SRUS), and there are very few reports on AHRU in children. SRUS is characterized by defecation coordination disorders such as chronic constipation and straining and is more common in teenagers.4 In SRUS, the rectal mucosal tissue usually exhibits characteristics such as fibrotic changes. In contrast, many patients with AHRU are bedridden older adults, and it has been reported that the rectal mucosal blood flow is reduced in the supine position.3 Pathological findings of the rectal mucosa show mild inflammation without fibrotic changes. Additionally, since most patients exhibit hypoalbuminemia, it is possible that malnutrition contributes to mucosal damage and delayed healing.5 Thus, nutritional support should be considered an important aspect of care in patients with AHRU because malnutrition in bedridden children is a risk factor for AHRU. The patient in our case was a severely disabled child in the same situation as a bedridden older adult, and sufficient nutritional management effectively improved the condition. Based on these characteristics, we believe this case is more consistent with AHRU rather than SRUS. This case report suggests that children with severe motor and intellectual disabilities, who are often bedridden and malnourished, are at risk of AHRU. It also suggests that nutritional management is important for AHRU treatment. Through this detailed exploration, we aimed to raise awareness of AHRU and promote further investigation into its occurrence in children, and ultimately improve diagnosis and treatment options, such as nutritional management, for those affected. Further research is warranted to better understand the occurrence, diagnosis, and management of AHRU in children with complex medical needs. K.I. conceived and designed the study and wrote the manuscript; T.D., H.N., A.M., and T.E. collected the data and revised the manuscript; H.T. revised the manuscript. All authors have read and approved the final manuscript. We would like to thank the members of the University of Tsukuba Hospital for their assistance. The authors declare no conflict of interest. The ethics committee of the University of Tsukuba approved this study. Written informed consent was obtained from the participants. The data that support the findings of this study are available upon request from the corresponding author. The data are not publicly available due to privacy or ethical restrictions.
Imagawa et al. (Thu,) studied this question.