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Case Reports
. 2023 Jan-Dec:11:23247096231179451.
doi: 10.1177/23247096231179451.

Clinical and Endoscopic Response to Anti-Tumor Necrosis Factor-Alpha Antibody Therapy in a Patient With Cronkhite-Canada Syndrome

Roham Salman Roghani  1 , Joline De Castro  1 , Adewale B Ajumobi  1   2
Affiliations
Case Reports

Clinical and Endoscopic Response to Anti-Tumor Necrosis Factor-Alpha Antibody Therapy in a Patient With Cronkhite-Canada Syndrome

Roham Salman Roghani et al. J Investig Med High Impact Case Rep. 2023 Jan-Dec.

Abstract

Cronkhite-Canada syndrome (CCS) is an acquired polyposis syndrome with gastrointestinal and extraintestinal manifestations. Given its rarity and lack of standard treatment, diagnosis and treatment are challenging. Steroid therapy and nutritional support are conventional treatments. There is no consensus on management of steroid-refractory cases. Here, we report the diagnosis and treatment course of a 54-year-old Asian male with CCS, whose initial treatment with prednisone 60 mg a day led to partial response and disease flare up during prednisone tapering. The use of infliximab and azathioprine led to promising remission of his symptoms.

Keywords: Cronkhite-Canada syndrome; cutaneous hyperpigmentation; gastroenterology; gastrointestinal polyps; onychodystrophy; steroid-resistant Cronkhite-Canada syndrome.

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Conflict of interest statement

The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Figures

Figure 1.
Figure 1.
Intestinal and extraintestinal clinical manifestation of the patient at presentation: (A) alopecia, hyperpigmentation of skin on the palms and scalp, and onychodystrophy in all digits most notably in thumbs. Multiple polyps accompanying mucosal edema and hyperemia were observed in the (B) stomach and (C) colon.
Figure 2.
Figure 2.
Follow-up endoscopic evaluation after treating with infliximab: Significant regression in both polyps’ size and number in the (A) stomach and (B) entire colon.
See this image and copyright information in PMC

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