Adenocarcinoma arising from a long-standing solitary rectal ulcer syndrome

Solitary rectal ulcer syndrome (SRUS) is a rare chronic disease characterized by a combination of clinical symptoms, endoscopic appearance, and histology findings, with periods of remission and relapse [1, 2]. It most commonly affects young adults [3]. Patients usually pass mucus and blood while defecating, but can also complain about tenesmus, straining on defecation, incontinence, or a sensation of incomplete evacuation [4]. SRUS is a benign condition, but some rare cases of associated adenocarcinomas have been described [5]. We report here the case of a 57-year-old woman with a history of a long-standing SRUS refractory to two surgical operations. After the second operation, a polypoid lesion appeared which was consistent with a cicatricial lesion. Five years later, endoscopic reevaluation revealed that the lesion had increased in size, and low-grade dysplasia was shown on biopsies. The patient was therefore sent to our endoscopy unit for resection of this lesion (▶Video 1). Endoscopy revealed a Paris 0-Is lesion; its base presented an adenomatous appearance, but in other parts there were some areas of irregular pit pattern (Kudo Vn) and vascular pattern (Sano IIIb) that could be consistent with adenocarcinoma or an inflammatory lesion (▶Fig. 1). Because of this appearance, en bloc resection of the lesion was decided upon, using a hybrid resection technique. Circumferential incision of the mucosa was performed around the lesion (▶Fig. 2). Then, the hot snare was placed into the incision and the lesion was removed (▶Fig. 3). A residual part was removed with the hot snare. The pathology report showed moderately differentiated adenocarcinoma infiltrating the submucosa by more than 5000μm within an inflammatory stroma. The resection was not R0 and the patient underwent radiotherapy and proctectomy. This case highlights the fact that we should be careful when we encounter a lesion in the setting of long-standing SRUS, and should consider resection.