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Case Reports
. 2025 Mar 30:46:e00706.
doi: 10.1016/j.crwh.2025.e00706. eCollection 2025 Jun.

Infliximab treatment for Cronkhite-Canada syndrome in pregnancy: A case report

Ayano Rosemary Nakamura  1 , Shuji Yamamoto  2 , Yoshitsugu Chigusa  1 , Masaki Mandai  1 , Haruta Mogami  1
Affiliations
Case Reports

Infliximab treatment for Cronkhite-Canada syndrome in pregnancy: A case report

Ayano Rosemary Nakamura et al. Case Rep Womens Health. .

Abstract

Cronkhite-Canada syndrome (CCS) is a rare nonhereditary disorder characterized by gastrointestinal polyps and protein-losing enteropathy. While an increasing number of CCS cases have been reported worldwide, no documented cases involving pregnant patients could be found. Consequently, optimal management strategies for CCS during the preconception period and pregnancy remain unclear., The present report concerns the case of a 36-year-old woman with steroid-refractory CCS stabilized with gastrointestinal surgeries and infliximab, an anti-tumor necrosis factor-α (TNF-α) agent, who became pregnant. Infliximab was continued throughout pregnancy and postpartum. Despite persistent hypoalbuminemia, sh delivered a healthy infant weighing 2518 g vaginally at 38 weeks and 2 days without CCS exacerbation. Both the patient and her infant experienced an uneventful postpartum course. This case suggests that maintaining disease control with anti-TNF-α therapy in pregnant patients with CCS may contribute to optimizing maternal and neonatal outcomes.

Keywords: Anti-TNFα therapy; Cronkhite-Canada syndrome; Hypoalbuminemia; Infliximab; Pregnancy.

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Figures

Fig. 1
Fig. 1
Diagnostic features of Cronkhite-Canada syndrome. (a) Characteristic nail dystrophy showing atrophy and onycholysis. (b) Skin hyperpigmentation on the patient's hand (arrows). (c, f) Endoscopic images showing multiple polyps in the stomach and colon, respectively. (d, g) Macroscopic findings of the resected specimens from distal gastrectomy and partial colectomy, showing numerous polyps. (e, h) Microscopic findings of polyps in stomach and colon showing glandular structures with variable dilatation, accompanied by edematous changes in the stroma and inflammatory cell infiltration. Hematoxylin and eosin stain, magnification ×100, scale bars = 200 μm.
Fig. 2
Fig. 2
Treatment course and changes in hemoglobin and serum albumin levels. PSL: prednisolone.
Fig. 3
Fig. 3
Abdominal computed tomography images (a) 21 days and (b) 121 days after initiation of infliximab. Multiple mass shadows suspected to be ileal and colonic polyps (arrowheads) show reduction in size.
Fig. 4
Fig. 4
Changes in hemoglobin and serum albumin levels during pregnancy and the peripartum period.
See this image and copyright information in PMC

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