Massa Roberto, Bruno Claudio, Martorana Alessandro, de Stefano Nicola, van Diggelen Otto P, Federico Antonio
Dipartimento di Neuroscienze, Università di Roma Tor Vergata and Fondazione Santa Lucia, Roma, Italy.
Muscle Nerve. 2008 Apr;37(4):530-6. doi: 10.1002/mus.20916.
Adult polyglucosan body disease (APBD) is characterized by the accumulation of insoluble glucose polymers within the central and peripheral nervous systems. A common missense mutation in the glycogen branching enzyme (GBE1) gene has been identified in Ashkenazi patients with APBD. We report on a non-Jewish patient with APBD on whom we performed proton magnetic resonance spectroscopic imaging of the brain. GBE activity in fibroblasts was markedly reduced, and a novel heterozygous mutation was identified in the GBE1 gene. Our findings widen the spectrum of APBD genotypes, underline the importance of performing GBE analysis in all APBD patients, and suggest that brain white matter degeneration in APBD may result from tissue damage involving axons and myelin.
成人多聚葡萄糖体病(APBD)的特征是在中枢和周围神经系统中积累不溶性葡萄糖聚合物。在患有APBD的阿什肯纳兹患者中已鉴定出糖原分支酶(GBE1)基因的常见错义突变。我们报告了一名患有APBD的非犹太患者,我们对其进行了脑部质子磁共振波谱成像。成纤维细胞中的GBE活性明显降低,并且在GBE1基因中鉴定出一种新的杂合突变。我们的研究结果拓宽了APBD基因型的范围,强调了对所有APBD患者进行GBE分析的重要性,并表明APBD中的脑白质变性可能是由涉及轴突和髓鞘的组织损伤引起的。