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胶质纤维酸性蛋白星形胶质细胞病在 DNAJC30 突变致 Leber 遗传性视神经病变患者中的共同发生。

Co-occurrence of glial fibrillary acidic protein astrocytopathy in a patient with Leber's hereditary optic neuropathy due to DNAJC30 mutations.

机构信息

IRCCS Istituto delle Scienze Neurologiche di Bologna, Bologna, Italy.

Dipartimento di Scienze Biomediche e Neuromotorie, Università di Bologna, Bologna, Italy.

出版信息

Eur J Neurol. 2024 Sep;31(9):e16344. doi: 10.1111/ene.16344. Epub 2024 May 17.

DOI:10.1111/ene.16344
PMID:38757769
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11295152/
Abstract

Leber's hereditary optic neuropathy (LHON) is a mitochondrial disease characterized by visual loss, and rarely associated with extraocular manifestations including multiple sclerosis-like lesions. The association of LHON and neuromyelitis optica spectrum disorders has rarely been reported. Here is reported a case of glial fibrillary acidic protein astrocytopathy presenting with area postrema syndrome in a patient with previously diagnosed recessive LHON due to mutations in the nuclear gene DNAJC30. This case emphasizes the necessity of extensive investigations for other treatable conditions in patients with LHON and otherwise unexplained extraocular involvement and the possibility that also visual symptoms can respond to immune therapy.

摘要

Leber 遗传性视神经病变(LHON)是一种以视力丧失为特征的线粒体疾病,很少伴有眼外表现,包括多发性硬化样病变。LHON 与视神经脊髓炎谱系疾病的关联很少有报道。本文报道了一例因核基因 DNAJC30 突变导致的隐性 LHON 患者出现胶质纤维酸性蛋白星形胶质细胞病,并伴有后区综合征。该病例强调了在 LHON 患者和其他原因不明的眼外受累患者中,对其他可治疗疾病进行广泛检查的必要性,以及视觉症状也可能对免疫治疗有反应的可能性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e42e/11295152/4c84383084b2/ENE-31-e16344-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e42e/11295152/4c84383084b2/ENE-31-e16344-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e42e/11295152/4c84383084b2/ENE-31-e16344-g001.jpg

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本文引用的文献

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Area Postrema Syndrome in Autoimmune Glial Fibrillary Acidic Protein Astrocytopathy: A Case Series and Literature Review.自身免疫性神经丝酸性蛋白星形胶质细胞病中的后区综合征:病例系列及文献复习。
Neurol Neuroimmunol Neuroinflamm. 2022 Sep 26;9(6). doi: 10.1212/NXI.0000000000200029. Print 2022 Nov.
2
The immune system as a driver of mitochondrial disease pathogenesis: a review of evidence.免疫系统作为线粒体疾病发病机制的驱动因素:证据综述。
Orphanet J Rare Dis. 2022 Sep 2;17(1):335. doi: 10.1186/s13023-022-02495-3.
3
DNAJC30 defect: a frequent cause of recessive Leber hereditary optic neuropathy and Leigh syndrome.
DNAJC30缺陷:隐性Leber遗传性视神经病变和Leigh综合征的常见病因。
Brain. 2022 Jun 3;145(5):1624-1631. doi: 10.1093/brain/awac052.
4
Late-onset Leber's hereditary optic neuropathy presenting with longitudinally extensive myelitis harbouring the m.14484T>C mutation: Extending the genotype-phenotype spectrum.伴有m.14484T>C突变的纵向广泛脊髓炎表现的迟发性Leber遗传性视神经病变:扩展基因型-表型谱
Mult Scler Relat Disord. 2021 Feb;48:102688. doi: 10.1016/j.msard.2020.102688. Epub 2020 Dec 15.
5
Mitochondrial DNA in inflammation and immunity.线粒体 DNA 与炎症和免疫。
EMBO Rep. 2020 Apr 3;21(4):e49799. doi: 10.15252/embr.201949799. Epub 2020 Mar 23.