Kiriştioğlu I, Balkan E, Kiliç N, Doğruyol H
Department of Pediatric Surgery, Uludağ University Faculty of Medicine, Bursa, Turkey.
Turk J Pediatr. 2000 Jan-Mar;42(1):56-60.
The solitary rectal ulcer syndrome (SRUS) is an unusual disorder in childhood. Although well recognized in adult literature, the pediatric experience with this condition is limited, so SRUS often goes unrecognized or misdiagnosed. There are very few pediatric case reports in the English literature. This report describes four patients who presented with rectal bleeding, constipation, mucous discharge, and lower abdominal pain, with a diagnosis of SRUS. The diagnosis was made by rectoscopy, defecogram, anorectal manometry and histopathological evaluation. In two patients, defecogram showed a rectocele with both, the sphincter failed to relax to voluntary squeeze pressure on anorectal manometric examination. The histopathological finding in all patients was fibrous obliteration of the lamina propria with disorientation of muscle fibers. All of the patients responded well to conservative therapy, which included defecation training, laxatives, sulfasalazine, and application of rectal sucralfate enema, and remained asymptomatic on the follow-up. Although rare in the pediatric population, SRUS should be relatively easy to recognize in the child with rectal bleeding, after elimination of other causes. If suspected, the diagnosis of SRUS may be made at endoscopy and confirmed by rectal biopsy.
孤立性直肠溃疡综合征(SRUS)在儿童期是一种罕见的疾病。尽管在成人文献中已得到充分认识,但儿科对此病症的经验有限,因此SRUS常常未被识别或误诊。英文文献中儿科病例报告极少。本报告描述了4例出现直肠出血、便秘、黏液便及下腹痛的患者,诊断为SRUS。诊断通过直肠镜检查、排粪造影、肛门直肠测压及组织病理学评估做出。2例患者的排粪造影显示直肠膨出,且在肛门直肠测压检查中,括约肌对自主挤压压力未能松弛。所有患者的组织病理学表现均为固有层纤维性闭塞伴肌纤维排列紊乱。所有患者对包括排便训练、泻药、柳氮磺胺吡啶及直肠硫糖铝灌肠在内的保守治疗反应良好,随访期间均无症状。尽管SRUS在儿科人群中罕见,但在排除其他病因后,对于有直肠出血的儿童应相对容易识别。若怀疑该病,可在内镜检查时做出SRUS的诊断,并通过直肠活检加以证实。