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

Abstract

Rationale: 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.

Patient concerns: 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.

Diagnoses: These clinical features and endoscopic findings were consistent with CCS which associated with perianal condyloma acuminatum with malignant transformation.

Intervention: Clinical remission was achieved with glucocorticoid, azathioprine, and nutritional support.

Outcome: 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.

Lessons: 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.

Figure 1

Perianal neoplasms as seen on physical examination.

Figure 2

Upper gastrointestinal endoscopy: the polyps in the gastric antrum show a carpet-like distribution.

Figure 3

Colonoscopy: the polyps in the ileocecal junction are distributed in clusters.

Figure 4

Capsule endoscopy: scattered polyps are seen in the jejunum.

Figure 5

Capsule endoscopy: the intestinal villi are hyperplastic, prolonged, and demonstrate a seaweed-like pattern.

Figure 6

Histopathology examination of Perianal neoplasms (hematoxylin-eosin stain, ×10).