University Teaching Hospital Neurology Research Office, Lusaka, Zambia.
University Teaching Hospital Neurology Research Office, Lusaka, Zambia; Department of Neurology, University of Rochester Medical Center, Rochester, New York.
Pediatr Neurol. 2023 Nov;148:173-177. doi: 10.1016/j.pediatrneurol.2023.08.038. Epub 2023 Aug 31.
Medical and rehabilitative advances increasingly transform management of rare genetic neuromuscular diseases (GNMDs) for children in the global north. Lack of information about GNMDs and related health care needs in sub-Saharan Africa threatens to widen pre-existing health disparities.
This is a cross-sectional study of probands enrolling in a study of GNMDs at the University Teaching Hospital in Lusaka, Zambia, a member of the International Consortium for Genomic Medicine in Neuromuscular Disease. Probands/caregivers were interviewed about utilization of medical, rehabilitative, and other support services by a research assistant. A neuromuscular neurologist and/or physiotherapist examined each case and completed an independent questionnaire regarding health service utilization for each proband. Diagnoses were made on available clinical and electrophysiologic data. Molecular findings were unavailable at the time of this analysis.
Among 50 probands, 52% were male with median age 12 (absolute range 2 months to 54 years). Motor neuron diseases (n = 16; 32%), muscle disorders (n = 20; 40%), and inherited polyneuropathies (n = 5; 10%) were most common. Six (15%) cases had insufficient clinical data to classify the GNMDs. Outside of primary care, patient/caregiver-reported access to recommended health services (n = 34; 69%) was challenging. Large disparities in current utilization of health care services versus clinician-recommended services are reported.
Paradigms to improve access to diagnostics and therapeutic interventions are needed for GNMDs in Zambia. Multidisciplinary clinics may improve access and utilization of needed health services. Qualitative and other research focused on improving referrals, access, and quality of available health services are greatly needed.
医学和康复方面的进步日益改变了北温带国家儿童罕见遗传性神经肌肉疾病(GNMD)的治疗方式。撒哈拉以南非洲地区缺乏有关 GNMD 及相关医疗保健需求的信息,这有可能扩大已存在的健康差距。
这是一项在赞比亚卢萨卡大学教学医院进行的 GNMD 研究中的先证者的横断面研究,该医院是国际神经肌肉疾病基因组医学联盟的成员。研究助理对先证者/照顾者进行了关于医疗、康复和其他支持服务利用情况的访谈。神经肌肉科医生和/或物理治疗师对每位病例进行了检查,并为每位先证者完成了一份关于卫生服务利用情况的独立问卷。根据现有的临床和电生理数据做出诊断。在进行这项分析时,分子检测结果尚不可用。
在 50 名先证者中,52%为男性,中位年龄为 12 岁(绝对范围为 2 个月至 54 岁)。运动神经元疾病(n=16;32%)、肌肉疾病(n=20;40%)和遗传性周围神经病(n=5;10%)最为常见。有 6 例(15%)病例的 GNMD 分类因临床资料不足而无法明确。除了初级保健之外,患者/照顾者报告难以获得推荐的卫生服务(n=34;69%)。卫生保健服务的实际利用率与临床医生推荐的利用率之间存在巨大差异。
赞比亚需要改善 GNMD 诊断和治疗干预的获取途径。多学科诊所可能会改善对所需卫生服务的获取和利用。非常需要开展定性和其他研究,以改善转介、获取和现有卫生服务的质量。