两兄妹合并中枢和外周脱髓鞘,是免疫介导还是遗传?

IF 2.4 Q2 CLINICAL NEUROLOGY PRACTICAL NEUROLOGY Pub Date : 2024-09-13 DOI:10.1136/pn-2024-004114
Kaminie Moodley, Anandan A Moodley, Stephanie Efthymiou, Henry Houlden, Pierre L A Bill, Vinod B Patel, Simon Rinaldi
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引用次数: 0

摘要

我们报告了与妊娠有关的两兄妹合并中枢和外周脱髓鞘的罕见病例,他们均表现为进行性四肢瘫痪和颅神经麻痹。姐姐的病程为复发-缓解,伴有视神经功能障碍,在复发期间死于呼吸功能不全。妹妹表现为定向障碍以及急性发作的肢体和面部无力。她对皮质类固醇治疗反应良好。他们的临床表现、对免疫调节疗法的反应、神经传导研究、脑脊液和组织学均支持后天性脱髓鞘病因。全基因组测序发现了两个基因的变异,而这两个基因以前从未与这种临床表型联系在一起。抗体介导的脱髓鞘血清学检测呈阴性。尽管发病机制尚不明确,但这些病例为探索获得性脱髓鞘的遗传、免疫和环境因素的融合提供了一个平台。我们将从神经免疫学和神经遗传学的角度讨论此类病例的鉴别诊断和诊断方法。
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Combined central and peripheral demyelination in two siblings, immune mediated or genetic?

We report unusual cases of combined central and peripheral demyelination in two siblings related to pregnancy, each presenting with progressive tetraparesis and cranial nerve palsies. The elder sister had a relapsing-remitting course with optic nerve dysfunction and died during a relapse from respiratory insufficiency. The younger sister presented with disorientation and acute-onset limb and facial weakness. She responded well to corticosteroid therapy. Their clinical presentation, response to immunomodulatory therapy, nerve conduction studies, cerebrospinal fluid and histology supported an acquired demyelinating cause. Whole-exome sequencing identified variants in two genes not previously linked to this clinical phenotype. Serological tests for antibody-mediated demyelination were negative. Despite the undefined pathogenesis, these cases provide a platform to explore the confluence of genetic, immune and environmental factors in the context of acquired demyelination. We discuss the differential diagnosis and a diagnostic approach to such cases from the perspectives of neuroimmunology and neurogenetics.

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来源期刊
PRACTICAL NEUROLOGY
PRACTICAL NEUROLOGY Medicine-Neurology (clinical)
CiteScore
3.70
自引率
3.60%
发文量
113
期刊介绍: The essential point of Practical Neurology is that it is practical in the sense of being useful for everyone who sees neurological patients and who wants to keep up to date, and safe, in managing them. In other words this is a journal for jobbing neurologists - which most of us are for at least part of our time - who plough through the tension headaches and funny turns week in and week out. Primary research literature potentially relevant to routine clinical practice is far too much for any neurologist to read, let alone understand, critically appraise and assimilate. Therefore, if research is to influence clinical practice appropriately and quickly it has to be digested and provided to neurologists in an informative and convenient way.
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