Division of Neuromuscular Diseases, Department of Neurology and Neurosurgery, Federal University of São Paulo (UNIFESP), São Paulo, SP, Brazil.
Department of Neurology, Columbia University Medical Center, New York, New York, USA.
J Inherit Metab Dis. 2021 May;44(3):534-543. doi: 10.1002/jimd.12325. Epub 2020 Nov 13.
Adult polyglucosan body disease (APBD) represents a complex autosomal recessive inherited neurometabolic disorder due to homozygous or compound heterozygous pathogenic variants in GBE1 gene, resulting in deficiency of glycogen-branching enzyme and secondary storage of glycogen in the form of polyglucosan bodies, involving the skeletal muscle, diaphragm, peripheral nerve (including autonomic fibers), brain white matter, spinal cord, nerve roots, cerebellum, brainstem and to a lesser extent heart, lung, kidney, and liver cells. The diversity of new clinical presentations regarding neuromuscular involvement is astonishing and transformed APBD in a key differential diagnosis of completely different clinical conditions, including axonal and demyelinating sensorimotor polyneuropathy, progressive spastic paraparesis, motor neuronopathy presentations, autonomic disturbances, leukodystrophies or even pure myopathic involvement with limb-girdle pattern of weakness. This review article aims to summarize the main clinical, biochemical, genetic, and diagnostic aspects regarding APBD with special focus on neuromuscular presentations.
成人多聚糖体病(APBD)是一种复杂的常染色体隐性遗传性神经代谢疾病,由于 GBE1 基因的纯合子或复合杂合致病性变异,导致糖原分支酶缺乏,糖原以多聚糖体的形式储存,累及骨骼肌、膈肌、周围神经(包括自主纤维)、脑白质、脊髓、神经根、小脑、脑干,在较小程度上还累及心脏、肺、肾和肝细胞。神经肌肉受累的新临床表现的多样性令人惊讶,将 APBD 转变为完全不同临床情况的关键鉴别诊断,包括轴索性和脱髓鞘感觉运动性多发性神经病、进行性痉挛性截瘫、运动神经元病表现、自主神经紊乱、白质营养不良,甚至纯肌病性肌无力伴肢带型模式。本文综述了 APBD 的主要临床、生化、遗传和诊断方面,特别关注神经肌肉表现。