一例膜性肾病并发克朗凯特-加拿大综合征的病例,用米佐利宾治疗成功。

IF 1 Q4 UROLOGY & NEPHROLOGY CEN Case Reports Pub Date : 2024-07-02 DOI:10.1007/s13730-024-00908-9
Hiroyuki Nakanoh, Kenji Tsuji, Shiho Morimoto, Kazuhiko Fukushima, Masaya Iwamuro, Haruhito A Uchida, Jun Wada
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引用次数: 0

摘要

克朗凯特-加拿大综合征(Cronkhite-Canada Syndrome,CCS)是一种非遗传性疾病,以非肿瘤性火腿肠息肉病、脱发、指甲萎缩、色素沉着和腹泻为特征。虽然 CCS 与肾炎之间的关系尚不清楚,但迄今为止已有 7 例 CCS 并发肾炎的报道,所有病例均为膜性肾病(MN)。一名 57 岁男子出现味觉障碍、脱发、甲板萎缩、皮肤色素沉着和频繁腹泻。内镜检查结果显示胃和大肠多发性息肉。鉴于上述情况,他被诊断为 CCS。泼尼松龙治疗后症状逐渐改善,但在泼尼松龙逐渐减量期间出现了尿蛋白和低蛋白血症。通过肾活检,他被诊断为 MN,免疫荧光显微镜下的 IgG 亚类染色显示,主要是弥漫性颗粒状毛细血管壁 IgG4 染色。继发性 MN 的病因未找到,包括恶性肿瘤。尽管接受了泼尼松龙和环孢素治疗,但肾病范围的蛋白尿仍持续存在。米佐利宾的额外治疗导致肾病综合征 1 型不完全缓解,这表明米佐利宾可能是类固醇耐药的 CCS MN 患者的一种治疗选择。考虑到慢性腹泻和蛋白丢失性肠病导致的低蛋白血症在 CCS 患者中发病率很高,蛋白尿可能会被忽视;因此,建议对 CCS 患者进行随访尿检。
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A case of membranous nephropathy complicated by Cronkhite-Canada syndrome successfully treated with mizoribine.

Cronkhite-Canada syndrome (CCS) is a non-hereditary disorder characterized by non-neoplastic hamartomatous gastrointestinal polyposis, hair loss, nail atrophy, hyperpigmentation, and diarrhea. While the relationship between CCS and nephritis remains unclear, seven cases of nephritis complicated by CCS have been reported to date, all of which were membranous nephropathy (MN). A 57-year-old man presented with taste disturbance, hair loss, nail plate atrophy, skin pigmentation, and frequent diarrhea. Endoscopic findings showed multiple polyposis of the stomach and large intestine. Given the above, he was diagnosed with CCS. The symptoms gradually improved with prednisolone treatment, although urinary protein and hypoproteinemia appeared during the tapering of prednisolone. He was diagnosed with MN using a renal biopsy, and immunofluorescence microscopy with IgG subclass staining showed predominantly diffuse granular capillary wall staining of IgG4. The cause of secondary MN was not found, including malignant tumors. Nephrotic-range proteinuria persisted despite treatment with prednisolone and cyclosporine. Additional treatment with mizoribine resulted in incomplete remission type 1 of nephrotic syndrome, suggesting that mizoribine may be a treatment option for patients with CCS with steroid-resistant MN. Considering a high prevalence of hypoproteinemia due to chronic diarrhea and protein-losing enteropathy in patients with CCS, proteinuria might be overlooked; thus, follow-up urinalysis would be recommended in patients with CCS.

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来源期刊
CEN Case Reports
CEN Case Reports UROLOGY & NEPHROLOGY-
CiteScore
1.90
自引率
0.00%
发文量
80
期刊介绍: Clinical and Experimental Nephrology (CEN) Case Reports is a peer-reviewed online-only journal, officially published biannually by the Japanese Society of Nephrology (JSN).  The journal publishes original case reports in nephrology and related areas.  The purpose of CEN Case Reports is to provide clinicians and researchers with a forum in which to disseminate their personal experience to a wide readership and to review interesting cases encountered by colleagues all over the world, from whom contributions are welcomed.
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