• Wei Wang, Xian-Yong Cheng, Feng Xue, Hai-Yan Liu, Hai-Feng Lian, and Cheng-Xia Liu.
• Binzhou Medical University Hospital, The Yellow Rive Second Road, Binzhou City, Shandong Province, China.
• Medicine (Baltimore). 2021 Mar 12; 100 (10): e25067e25067.

RationaleCronkhite-Canada syndrome (CCS) is a rare non-familial polyposis syndrome characterized by multiple gastrointestinal polyps with the ectodermal triad. To date, many complications of CCS have been reported in the literature, but perianal condyloma acuminatum with malignant transformation has not been included.Patient ConcernsThis report presents the case of a 52-year-old Chinese man who presented with diarrhea, loss of appetite, and weight loss. He developed skin pigmentation and atrophy of the fingernails and toenails. Upper gastrointestinal endoscopy, colonoscopy, capsule endoscopy, and enteroscopy revealed diffuse polyps along the entire digestive tract. Histopathological examination revealed polyps of different pathological types dominated by hamartoma. Physical examination revealed a crissum cauliflower-like neoplasm (2.5 × 2.0 cm). After perianal tumor resection, pathology suggested that this was a perianal condylomatous lesion with malignant transformation, as well as well-differentiated squamous cell carcinoma.DiagnosesThese clinical features and endoscopic findings were consistent with CCS which associated with perianal condyloma acuminatum with malignant transformation.InterventionClinical remission was achieved with glucocorticoid, azathioprine, and nutritional support.OutcomeAt the 4-year follow-up, the patient had no diarrhea or loss of appetite, had gained 13 kg in weight, and the perianal tumor had not recurred.LessonsNo previous report has described CCS in a patient with perianal condyloma acuminatum with malignant transformation. As both conditions are related to immune disorders, their occurrence may be correlated.Copyright © 2021 the Author(s). Published by Wolters Kluwer Health, Inc.

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