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丹侬病继发垂头综合征:一例报告

Dropped Head Syndrome Secondary to Danon Disease: A Case Report.

作者信息

Bhat Vivek, Harikrishna Ganaraja V, Kumar Hyndav, Kodapala Suresha

机构信息

Internal Medicine, St. John's Medical College, Bangalore, IND.

Neurology, Vydehi Institute of Medical Sciences & Research Centre, Bangalore, IND.

出版信息

Cureus. 2023 Jun 30;15(6):e41191. doi: 10.7759/cureus.41191. eCollection 2023 Jun.

Abstract

Dropped head syndrome (DHS) is characterized by neck extensor muscle weakness, which may be isolated or secondary to another neurologic diagnosis. DHS, due to lysosomal storage disorders, has not been reported in the literature. We present a 21-year-old male who had complaints of slowly worsening difficulty swallowing for the past eight years, along with difficulty keeping his head erect. His past medical history was significant for apical hypertrophic cardiomyopathy (HCM), and he had a history of sudden cardiac death in his immediate family. Clinical examination was significant only for neck extensor muscle weakness. His laboratory investigations were unremarkable, save for a significantly elevated creatine kinase (CK). Finally, whole exome sequencing identified a hemizygous stop gain variant in the lysosome-associated membrane protein 2 (LAMP-2) gene, pointing to a diagnosis of Danon disease (DD). DD is a rare, X-linked, inherited disease, due to a defect in the LAMP-2 gene that disrupts lysosomal autophagy. It is characterized by a triad of HCM, skeletal myopathy, and intellectual disability. Males typically suffer a more severe phenotype, and the cardiac disease drives its prognosis. Management involves regular cardiac monitoring, with appropriate physical therapy for myopathy and multidisciplinary treatment for intellectual disability. We suggest that DD be considered in the differential diagnosis for patients with HCM and elevated CK.

摘要

低头综合征(DHS)的特征是颈部伸肌肌无力,其可能是孤立性的,也可能继发于其他神经学诊断。文献中尚未报道过由溶酶体贮积症引起的DHS。我们报告一名21岁男性,他在过去八年中主诉吞咽困难逐渐加重,同时难以保持头部直立。他既往有肥厚型心肌病(HCM)病史,且直系亲属中有心脏性猝死病史。临床检查仅发现颈部伸肌肌无力。除肌酸激酶(CK)显著升高外,他的实验室检查无异常。最后,全外显子组测序在溶酶体相关膜蛋白2(LAMP-2)基因中发现了一个半合子终止获得变异,提示诊断为丹农病(DD)。DD是一种罕见的X连锁遗传病,由于LAMP-2基因缺陷导致溶酶体自噬功能障碍。其特征为HCM、骨骼肌病和智力残疾三联征。男性通常表现出更严重的表型,且心脏疾病决定其预后。治疗包括定期心脏监测,针对肌病进行适当的物理治疗,以及针对智力残疾进行多学科治疗。我们建议,对于患有HCM且CK升高的患者,鉴别诊断时应考虑DD。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/bae2/10387177/306821920583/cureus-0015-00000041191-i01.jpg

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