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英夫利昔单抗治疗妊娠期克朗凯特-加拿大综合征:一例报告。

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

作者信息

Nakamura Ayano Rosemary, Yamamoto Shuji, Chigusa Yoshitsugu, Mandai Masaki, Mogami Haruta

机构信息

Department of Gynecology and Obstetrics, Graduate School of Medicine, Kyoto University, 54 Shogoin Kawahara-cho, Sakyo-ku, Kyoto 606-8507, Japan.

Department of Gastroenterology and Hepatology, Graduate School of Medicine, Kyoto University, 54 Shogoin Kawahara-cho, Sakyo-ku, Kyoto 606-8507, Japan.

出版信息

Case Rep Womens Health. 2025 Mar 30;46:e00706. doi: 10.1016/j.crwh.2025.e00706. eCollection 2025 Jun.

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.

摘要

克朗凯特-加拿大综合征(CCS)是一种罕见的非遗传性疾病,其特征为胃肠道息肉和蛋白丢失性肠病。虽然全球范围内报告的CCS病例越来越多,但未发现有涉及孕妇的病例记录。因此,孕前和孕期CCS的最佳管理策略仍不明确。本报告涉及一名36岁患有类固醇难治性CCS的女性病例,该患者经胃肠道手术和抗肿瘤坏死因子-α(TNF-α)药物英夫利昔单抗治疗后病情稳定,随后怀孕。整个孕期及产后均持续使用英夫利昔单抗。尽管持续存在低白蛋白血症,但她在38周零2天时经阴道顺利分娩一名体重2518克的健康婴儿,且CCS未加重。患者及其婴儿产后过程均顺利。该病例表明,对患有CCS的孕妇采用抗TNF-α治疗维持疾病控制可能有助于优化母婴结局。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3b6d/11999527/5ac6b534201d/gr1.jpg

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本文引用的文献

1
Cronkhite-Canada syndrome: A case report and literature review.
Medicine (Baltimore). 2024 Oct 25;103(43):e40242. doi: 10.1097/MD.0000000000040242.
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Clinical and Endoscopic Response to Anti-Tumor Necrosis Factor-Alpha Antibody Therapy in a Patient With Cronkhite-Canada Syndrome.
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