Suppr超能文献

女性在脑腱黄瘤病中经历更严重的疾病过程。

Females experience a more severe disease course in Batten disease.

机构信息

University of Rochester, Rochester, NY, USA.

出版信息

J Inherit Metab Dis. 2012 May;35(3):549-55. doi: 10.1007/s10545-011-9421-6. Epub 2011 Dec 14.

Abstract

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.

摘要

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。

相似文献

1
Females experience a more severe disease course in Batten disease.
J Inherit Metab Dis. 2012 May;35(3):549-55. doi: 10.1007/s10545-011-9421-6. Epub 2011 Dec 14.
3
Do females with juvenile ceroid lipofuscinosis (Batten disease) have a more severe disease course? The Danish experience.
Eur J Paediatr Neurol. 2013 May;17(3):265-8. doi: 10.1016/j.ejpn.2012.10.011. Epub 2012 Nov 21.
4
The Unified Batten Disease Rating Scale (UBDRS): Validation and reliability in an independent CLN3 disease sample.
Eur J Paediatr Neurol. 2022 May;38:62-65. doi: 10.1016/j.ejpn.2022.03.005. Epub 2022 Apr 4.
5
A clinical rating scale for Batten disease: reliable and relevant for clinical trials.
Neurology. 2005 Jul 26;65(2):275-9. doi: 10.1212/01.wnl.0000169019.41332.8a.
6
Quantifying physical decline in juvenile neuronal ceroid lipofuscinosis (Batten disease).
Neurology. 2011 Nov 15;77(20):1801-7. doi: 10.1212/WNL.0b013e318237f649. Epub 2011 Oct 19.
8
Standardized assessment of seizures in patients with juvenile neuronal ceroid lipofuscinosis.
Dev Med Child Neurol. 2015 Apr;57(4):366-71. doi: 10.1111/dmcn.12634. Epub 2014 Nov 11.
9
Genotype does not predict severity of behavioural phenotype in juvenile neuronal ceroid lipofuscinosis (Batten disease).
Dev Med Child Neurol. 2010 Jul;52(7):637-43. doi: 10.1111/j.1469-8749.2010.03628.x. Epub 2010 Feb 19.

引用本文的文献

1
Impact of CLN3 Disease on Child Quality of Life and Family Function.
Pediatr Neurol. 2025 Jun 16;170:17-25. doi: 10.1016/j.pediatrneurol.2025.06.008.
2
Therapeutic antisense oligonucleotide mitigates retinal dysfunction in a pig model of CLN3 Batten disease.
bioRxiv. 2025 May 31:2025.05.30.656864. doi: 10.1101/2025.05.30.656864.
3
A recessive variant is responsible for delayed-onset retinal degeneration in Hereford cattle.
J Vet Diagn Invest. 2024 May;36(3):438-446. doi: 10.1177/10406387241239918. Epub 2024 Mar 22.
4
The parent and family impact of CLN3 disease: an observational survey-based study.
Orphanet J Rare Dis. 2024 Mar 18;19(1):125. doi: 10.1186/s13023-024-03119-8.
6
Overlap between ophthalmology and psychiatry - A narrative review focused on congenital and inherited conditions.
Psychiatry Res. 2024 Jan;331:115629. doi: 10.1016/j.psychres.2023.115629. Epub 2023 Nov 25.
7
Inborn Errors of Metabolism with Ataxia: Current and Future Treatment Options.
Cells. 2023 Sep 19;12(18):2314. doi: 10.3390/cells12182314.
8
Protracted CLN3 Batten disease in mice that genetically model an exon-skipping therapeutic approach.
Mol Ther Nucleic Acids. 2023 Jun 3;33:15-27. doi: 10.1016/j.omtn.2023.05.025. eCollection 2023 Sep 12.
9
A novel porcine model of CLN3 Batten disease recapitulates clinical phenotypes.
Dis Model Mech. 2023 Aug 1;16(8). doi: 10.1242/dmm.050038. Epub 2023 Aug 7.
10
Early recognition of CLN3 disease facilitated by visual electrophysiology and multimodal imaging.
Doc Ophthalmol. 2023 Jun;146(3):241-256. doi: 10.1007/s10633-023-09930-1. Epub 2023 Mar 25.

本文引用的文献

1
Quantifying physical decline in juvenile neuronal ceroid lipofuscinosis (Batten disease).
Neurology. 2011 Nov 15;77(20):1801-7. doi: 10.1212/WNL.0b013e318237f649. Epub 2011 Oct 19.
2
Immunosuppression alters disease severity in juvenile Batten disease mice.
J Neuroimmunol. 2011 Jan;230(1-2):169-72. doi: 10.1016/j.jneuroim.2010.08.024.
3
Estrogen actions in the brain and the basis for differential action in men and women: a case for sex-specific medicines.
Pharmacol Rev. 2010 Jun;62(2):155-98. doi: 10.1124/pr.109.002071. Epub 2010 Apr 14.
4
Genotype does not predict severity of behavioural phenotype in juvenile neuronal ceroid lipofuscinosis (Batten disease).
Dev Med Child Neurol. 2010 Jul;52(7):637-43. doi: 10.1111/j.1469-8749.2010.03628.x. Epub 2010 Feb 19.
5
Sexual dimorphism in autoimmune disease.
Curr Mol Med. 2009 Dec;9(9):1058-79. doi: 10.2174/156652409789839116.
6
Neuropsychological symptoms of juvenile-onset batten disease: experiences from 2 studies.
J Child Neurol. 2007 May;22(5):621-7. doi: 10.1177/0883073807302603.
7
Neurotrophic and neuroprotective actions of estrogen: basic mechanisms and clinical implications.
Steroids. 2007 May;72(5):381-405. doi: 10.1016/j.steroids.2007.02.003. Epub 2007 Feb 21.
10
A clinical rating scale for Batten disease: reliable and relevant for clinical trials.
Neurology. 2005 Jul 26;65(2):275-9. doi: 10.1212/01.wnl.0000169019.41332.8a.

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验