Department of Neurology, Boston Children's Hospital and Harvard Medical School, Boston, Massachusetts.
Department of Neurology, Boston Children's Hospital and Harvard Medical School, Boston, Massachusetts; Boston University Clinical Investigation Master's Program, Boston, Massachusetts.
Pediatr Neurol. 2023 Jan;138:71-80. doi: 10.1016/j.pediatrneurol.2022.10.006. Epub 2022 Oct 26.
Evidence of the impact of genetic diagnosis on medical management in individuals with previously unexplained epilepsy is lacking in the literature. Our goal was to determine the impact of genetic diagnosis on medical management in a cohort of individuals with early-onset epilepsy.
We performed detailed phenotyping of individuals with epilepsy who underwent clinical genetic testing with an epilepsy panel and/or exome sequencing at Boston Children's Hospital between 2012 and 2019. We assessed the impact of genetic diagnosis on medical management.
We identified a genetic etiology in 152 of 602 (25%) individuals with infantile- or childhood-onset epilepsy who underwent next-generation sequencing. Diagnosis impacted medical management in at least one category for 72% of patients (110 of 152) and in more than one category in 34%. Treatment was impacted in 45% of individuals, including 36% with impact on antiseizure medication choice, 7% on use of disease-specific vitamin or metabolic treatments, 3% on pathway-driven off-label use of medications, and 10% on discussion of gene-specific clinical trials. Care coordination was impacted in 48% of individuals. Counseling on a change in prognosis was reported in 28% of individuals, and 1% of individuals had a correction of diagnosis. Impact was documented in 13 of 13 individuals with neurotypical development and in 55% of those with epilepsy onset after age two years.
We demonstrated meaningful impact of genetic diagnosis on medical care and prognosis in over 70% of individuals, including those with neurotypical development and age of epilepsy onset after age two years.
目前文献中缺乏遗传诊断对既往不明原因癫痫患者医疗管理影响的证据。我们的目标是确定遗传诊断对早发性癫痫患者队列医疗管理的影响。
我们对 2012 年至 2019 年在波士顿儿童医院接受癫痫组套或外显子测序临床基因检测的癫痫患者进行了详细的表型分析。我们评估了遗传诊断对医疗管理的影响。
我们在 602 例婴儿期或儿童期起病的癫痫患者中发现了 152 例(25%)有遗传学病因。诊断对至少一个类别的 72%(110/152)患者的医疗管理产生影响,对 34%的患者产生了多个类别的影响。45%的患者治疗受到影响,包括 36%抗癫痫药物选择受到影响,7%使用疾病特异性维生素或代谢治疗,3%使用药物通路驱动的超说明书用药,10%讨论基因特异性临床试验。48%的患者受到照护协调的影响。28%的患者报告预后改变咨询,1%的患者修正了诊断。13 例神经认知正常发育的患者和 55%癫痫发病年龄大于 2 岁的患者均记录了影响。
我们证明了遗传诊断对 70%以上患者的医疗护理和预后有显著影响,包括神经认知正常发育和癫痫发病年龄大于 2 岁的患者。