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骨肉瘤患者接受异环磷酰胺治疗后出现伴有核肿大性间质性肾炎的范科尼综合征:一例报告

Fanconi syndrome with karyomegalic interstitial nephritis after ifosfamide treatment for osteosarcoma: a case report.

作者信息

Kita Yohei, Shirai Sayuri, Koyama Teppei, Makinouchi Ryuichiro, Machida Shinji, Matsui Katsuomi, Koike Junki, Imai Naohiko

机构信息

Division of Nephrology and Hypertension, Department of Internal Medicine, St. Marianna University Yokohama City Seibu Hospital, Yokohama, Japan.

Department of Nephrology, Shin-Yurigaoka General Hospital, Kawasaki, Japan.

出版信息

CEN Case Rep. 2025 Feb;14(1):65-70. doi: 10.1007/s13730-024-00907-w. Epub 2024 Jul 2.

Abstract

Patients with ifosfamide-induced renal damage present with Fanconi syndrome. Karyomegalic nephropathy/interstitial nephritis (KNIN) is a rare form of chronic tubulo-interstitial nephritis that was initially considered a type of familial nephropathy. However, several reports of drug-induced KNIN, i.e., KNIN-like nephropathy, have been reported in recent years. We present the case of an 18-year-old man who presented with Fanconi syndrome and progressive renal dysfunction after receiving chemotherapy including ifosfamide and cisplatin for right femoral osteosarcoma. Renal biopsy revealed numerous atrophied tubular epithelial cells with large, polymorphic nuclei, and the definitive diagnosis was KNIN. Most patients with KNIN-like nephropathy who receive ifosfamide are concomitantly treated with cisplatin, indicating that ifosfamide and cisplatin might act synergistically to increase the risk for KNIN-like nephropathy. Further investigation in case series is warranted to reveal potential treatment approaches and to evaluate prognosis.

摘要

异环磷酰胺诱导的肾损伤患者会出现范科尼综合征。核肿大性肾病/间质性肾炎(KNIN)是一种罕见的慢性肾小管间质性肾炎,最初被认为是一种家族性肾病。然而,近年来已有几例药物诱导的KNIN(即类KNIN肾病)的报道。我们报告了一例18岁男性患者,该患者在接受包括异环磷酰胺和顺铂的化疗治疗右股骨骨肉瘤后出现范科尼综合征和进行性肾功能不全。肾活检显示大量萎缩的肾小管上皮细胞,细胞核大且形态多样,最终诊断为KNIN。大多数接受异环磷酰胺治疗的类KNIN肾病患者同时接受顺铂治疗,这表明异环磷酰胺和顺铂可能协同作用增加类KNIN肾病的风险。有必要对病例系列进行进一步研究,以揭示潜在的治疗方法并评估预后。

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本文引用的文献

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Drug-induced renal Fanconi syndrome.药物诱导的肾性范可尼综合征。
QJM. 2014 Apr;107(4):261-9. doi: 10.1093/qjmed/hct258. Epub 2013 Dec 24.
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Ifosfamide induced Fanconi syndrome.异环磷酰胺诱发范科尼综合征。
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Karyomegalic-like nephropathy, Ewing's sarcoma and ifosfamide therapy.巨细胞样肾病变、尤文肉瘤和异环磷酰胺治疗。
Pediatr Nephrol. 2011 Jul;26(7):1163-6. doi: 10.1007/s00467-011-1815-z. Epub 2011 Mar 18.

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