Cronkhite-Canada syndrome associated with perianal condyloma acuminatum with malignant transformation: A case report

Cronkhite-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...

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Bibliographic Details
Published in:Medicine (Baltimore) 2021-03, Vol.100 (10), p.e25067
Main Authors: Wang, Wei, Cheng, Xian-Yong, Xue, Feng, Liu, Hai-Yan, Lian, Hai-Feng, Liu, Cheng-Xia
Format: Article
Language:English
Subjects:
Anal Canal - pathology
Anal Canal - surgery
Anus Neoplasms - diagnosis
Anus Neoplasms - etiology
Anus Neoplasms - pathology
Anus Neoplasms - therapy
Carcinoma, Squamous Cell - diagnosis
Carcinoma, Squamous Cell - etiology
Carcinoma, Squamous Cell - pathology
Carcinoma, Squamous Cell - therapy
Cell Transformation, Neoplastic
Clinical Case Report
Condylomata Acuminata - diagnosis
Condylomata Acuminata - etiology
Condylomata Acuminata - pathology
Condylomata Acuminata - therapy
Endoscopy, Gastrointestinal
Glucocorticoids - administration & dosage
Glutamine - administration & dosage
Humans
Intestinal Polyposis - complications
Intestinal Polyposis - diagnosis
Intestinal Polyposis - therapy
Male
Middle Aged
Nutritional Support
Treatment Outcome
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Summary:Cronkhite-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. This 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. These clinical features and endoscopic findings were consistent with CCS which associated with perianal condyloma acuminatum with malignant transformation. Clinical remission was achieved with glucocorticoid, azathioprine, and nutritional support. At 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. No 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.
ISSN:0025-7974
1536-5964
DOI:10.1097/MD.0000000000025067