一名线粒体 DNA 3243A>G 突变的青少年患者的肾脏和心脏活检结果:母亲肾移植后肾脏预后良好。

Clinical nephrology. Case studies Pub Date : 2024-06-25 eCollection Date: 2024-01-01 DOI:10.5414/CNCS111422
Hisashi Kamido, Shigekazu Kurihara, Yuki Oba, Masayuki Yamanouchi, Tatsuya Suwabe, Katsuyuki Miki, Yuki Nakamura, Yasuo Ishi, Kei Kono, Kenichi Ohashi, Naoki Sawa, Yoshifumi Ubara
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摘要

我们研究了一名 16 岁男性肾活检发现的局灶节段性肾小球硬化症周围变异的发病机制。该病对类固醇治疗无效,在第二次肾活检时,在荚膜细胞中新发现线粒体异常增殖。该患者还出现了迟发性听力损失,并有糖尿病家族史,基因检测证实其线粒体DNA突变为3243A>G(48%)。开始血液透析 8 个月后,可能由于快速脱水,患者出现了脑病。将透析改为连续不卧床腹膜透析后,脑病得到缓解,但患者出现了心肌肥厚,可能是因为心肌对充血反应过度。心肌活检显示心肌线粒体增生。在从他母亲那里移植了异型增殖率为 4% 的肾脏后,心肌病得到了改善,4 年来肾功能一直保持稳定。我们推测,肾脏和心脏的线粒体形态异常可能是线粒体遗传病的特征,对于预后不良的线粒体肾病,从异质性低的母亲处进行肾移植是可取的。
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Renal and cardiac biopsy findings in an adolescent patient with the 3243A>G mitochondrial DNA mutation: Favorable renal prognosis post renal transplantation from the mother.

We investigated the pathogenesis of a perihilar variant of focal segmental glomerulosclerosis detected by kidney biopsy in a 16-year-old male. The disease was refractory to steroid therapy, and at the second kidney biopsy, abnormal mitochondrial proliferation was newly observed in the podocytes. The patient also developed late-onset hearing loss and had a family history of diabetes, and genetic testing confirmed the mitochondrial DNA mutation 3243A>G (48%). Eight months after hemodialysis was started, encephalopathy occurred presumably due to rapid dehydration. After changing dialysis into continuous ambulatory peritoneal dialysis, encephalopathy was resolved, but the patient developed myocardial hypertrophy, probably because of the myocardial overreaction to congestion. A myocardial biopsy showed mitochondrial proliferation in the myocardium. After renal transplantation from his mother with a heteroplasmy of 4%, the cardiomyopathy improved, and the renal function has remained stable for 4 years. We speculated that the abnormal mitochondrial morphology in the kidney and heart may be characteristic of mitochondrial genetic disease, and renal transplantation from the mother with a low heteroplasmy was considered desirable for mitochondrial nephropathy with poor prognosis.

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