Montalbetti Lorenza, Soragna Debora, Ratti Maria Teresa, Bini Paola, Buscone Simona, Moglia Arrigo
Department of Neuroscience, University of Pavia, IRCCS "C. Mondino Institute of Neurology" Pavia, Italy.
Funct Neurol. 2004 Jul-Sep;19(3):171-9.
Nasu-Hakola disease (NHD, polycystic lipomembranous osteodysplasia with sclerosing leukoencephalopathy, PLOSL) is a recessively inherited disorder characterized by systemic bone cysts and progressive presenile dementia associated with sclerosing encephalopathy. The disease has a worldwide distribution, but most patients have been reported in Finland and in Japan; in Italy there are anecdotal reports. The combination of neuropsychiatric symptoms and bone cysts is unique to this disease, which we believe to be underestimated in Italy. The molecular defect has been identified in loss-of-function mutations in the TYROBP gene in Finnish and in Japanese patients, and in the TREM2 gene in other families of different ethnic origins. We reviewed the international literature to define better the diagnostic steps and to draw the attention of neurologists and orthopaedic specialists to the disease. The identification of new cases followed by appropriate genetic counselling, genetic analysis, and study of the territorial distribution of affected patients could be a good strategy to follow in order to improve understanding of the disease.
那须-哈科拉病(NHD,伴有硬化性白质脑病的多囊性脂膜性骨发育异常,PLOSL)是一种隐性遗传性疾病,其特征为全身性骨囊肿以及与硬化性脑病相关的进行性早老性痴呆。该疾病在全球范围内均有分布,但大多数患者报告来自芬兰和日本;在意大利仅有个别病例报告。神经精神症状与骨囊肿的组合是这种疾病所特有的,我们认为在意大利该疾病被低估了。在芬兰和日本患者中,已确定分子缺陷在于TYROBP基因的功能丧失突变,而在其他不同种族的家族中则是TREM2基因。我们查阅了国际文献,以更好地界定诊断步骤,并引起神经科医生和骨科专家对该疾病的关注。识别新病例,随后进行适当的遗传咨询、基因分析以及对患病患者地域分布的研究,可能是为增进对该疾病的了解而应遵循的良好策略。