Solitary rectal ulcer syndrome (SRUS) is a chronic infrequent rectal lesion, but SRUS canceration can be found in specific cases. Currently, there is no definite diagnosis for this anomaly. The following is the case of a 29-year-old male patient with long-standing SRUS, who rapidly developed progression to mucinous adenocarcinoma within 5 years. Colonoscopy and pathological examination confirmed the diagnosis of SRUS mucinous adenocarcinoma. Immunohistochemical analysis showed the positive expression in MLH1(+), MSH2(+), MSH6(+), PMS2(+), HER2(+). FOLFOX6 chemotherapy followed by surgery is effective for controlling SRUS-related mucinous adenocarcinoma progression, during the follow-up for the first 2 years. This article suggests that clinicians should pay close attention to canceration in patients with SRUS to improve patient outcomes.

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