Carlén B, Englund E
Department of Pathology and Cytology, University Hospital, Lund, Sweden.
Ultrastruct Pathol. 2001 Jul-Aug;25(4):285-8. doi: 10.1080/019131201753136296.
Neuronal ceroid lipofuscinoses (NCLs) represent a large group of inherited neurodegenerative disorders characterized by an abnormal accumulation of lipopigment in neuronal and extraneuronal cells. The authors present a case of juvenile neuronal ceroid lipofuscinosis in a 7-year-old boy. Ultrastructural examination of a skin biopsy disclosed deposits of curvilinear profiles and fingerprint-like structures in epithelial cells of sweat glands, endothelial cells, peripheral nerve endings, and fibroblasts. These findings allowed specific confirmation of the assumed diagnosis of juvenile neuronal ceroid lipofuscinosis. Due to the genotypic and phenotypic variability within the group of NCLs, the clinical investigation may be long and complicated. With the NCL disorders in mind, an accurate diagnosis based on ultrastructural examination of a skin biopsy may shorten this investigation, thus benefitting the patient.
神经元蜡样脂褐质沉积症(NCLs)是一大类遗传性神经退行性疾病,其特征是脂色素在神经元和神经外细胞中异常蓄积。作者报告了一例7岁男孩的青少年型神经元蜡样脂褐质沉积症。对皮肤活检组织进行超微结构检查发现,汗腺上皮细胞、内皮细胞、外周神经末梢和成纤维细胞中有曲线状轮廓和指纹样结构的沉积物。这些发现使得青少年型神经元蜡样脂褐质沉积症的假定诊断得到了特异性确认。由于NCLs组内存在基因型和表型变异,临床调查可能漫长且复杂。考虑到NCL疾病,基于皮肤活检组织超微结构检查的准确诊断可能会缩短这一调查过程,从而使患者受益。