University of Rochester, Rochester, NY, USA.
J Inherit Metab Dis. 2012 May;35(3):549-55. doi: 10.1007/s10545-011-9421-6. Epub 2011 Dec 14.
Juvenile neuronal ceroid lipofuscinosis (JNCL; CLN3 disease; Batten disease) is an autosomal recessive neurodegenerative disease of childhood. Symptoms typically present at school age with vision loss followed by progressive cognitive decline, motor dysfunction, seizures, and behavior problems. Studies on sex differences in JNCL have yielded mixed results, but parent anecdotes suggest that females experience a more precipitous disease course. Therefore, we sought to determine if sex-based differences exist in JNCL. We used data from the Unified Batten Disease Rating Scale (UBDRS), the Batten Disease Support and Research Association (BDSRA) database, and the PedsQL quality of life (QoL) survey to evaluate sex-based differences in functional independence and time from symptom onset to death. On average, females had JNCL symptom onset one year later and death one year earlier than did males. Despite a later age at onset, females had lower functional capability, earlier loss of independent function, and lower physical QoL. Future research in sex differences in JNCL may help to further understand the biological mechanisms underpinning the disease course and may point to targeted therapies.
青少年神经元蜡样脂褐质沉积症(JNCL;CLN3 病;Batten 病)是一种常染色体隐性遗传的儿童神经退行性疾病。症状通常在学龄期出现,表现为视力丧失,随后进行性认知能力下降、运动功能障碍、癫痫发作和行为问题。关于 JNCL 中性别差异的研究结果不一,但家长的轶事表明,女性的疾病进程更为急剧。因此,我们试图确定 JNCL 是否存在基于性别的差异。我们使用统一的 Batten 疾病评定量表(UBDRS)、Batten 疾病支持和研究协会(BDSRA)数据库以及 PedsQL 生活质量(QoL)调查的数据,评估了功能独立性以及从症状出现到死亡的时间方面的性别差异。平均而言,女性的 JNCL 症状发作比男性晚一年,死亡比男性早一年。尽管发病年龄较晚,但女性的功能能力较低,独立功能丧失较早,身体 QoL 较低。未来对 JNCL 中性别差异的研究可能有助于进一步了解疾病进程的生物学机制,并可能指向靶向治疗。