Bisma Aziz, Ahmed A Arif, Kulsum Kazi, Salman Kirmani, Zeeshan Ansar, Asghar Nasir, Shahnaz Hamid Ibrahim, Khairunnisa Mukhtiar Ahmed, Zahra Hasan, Sara Khan
{"title":"巴基斯坦脊髓性肌萎缩症和肌肉萎缩症登记的脊髓性肌萎缩症患者分析。","authors":"Bisma Aziz, Ahmed A Arif, Kulsum Kazi, Salman Kirmani, Zeeshan Ansar, Asghar Nasir, Shahnaz Hamid Ibrahim, Khairunnisa Mukhtiar Ahmed, Zahra Hasan, Sara Khan","doi":"10.1177/22143602241301657","DOIUrl":null,"url":null,"abstract":"<p><p>BackgroundSpinal Muscular Atrophy (SMA) leads to motor neuron loss, with progressive muscle weakness and wasting. Nationwide registries for neuromuscular diseases are pivotal for assessing epidemiology, preparing for clinical trials, and for adopting standardized management guidelines.ObjectivesThis paper aims to present data gathered during the establishment of Pakistan's inaugural registry for genetically confirmed SMA cases.MethodsIn this retrospective study, 215 participants with genetically confirmed SMA were recruited. Telephonic interviews were conducted to collect data for the Muscular Disease Registry of Pakistan that was analyzed using STATA version 17.0.ResultsSMA type 1 was the most common type (71.2%, n = 153). Amongst patients who were tested for survival motor neuron (<i>SMN2</i>) copies, the majority (84.4%, n = 168) had two <i>SMN2</i> copies. SMA types were significantly associated with the ability to sit (p < 0.001) and walk (p < 0.001), and usage of a wheelchair (p = 0.0054). <i>SMN2</i> copy numbers were significantly associated with the ability to sit (p = 0.020) and walk (p = 0.031).ConclusionsThis study highlights the high prevalence of SMA genotypes and phenotypes associated with severe disease in our population. Our findings reiterate the challenging prognosis for Pakistani children with SMA and underscore the necessity of the development of nationwide newborn screening programs and making treatments available.</p>","PeriodicalId":16536,"journal":{"name":"Journal of neuromuscular diseases","volume":" ","pages":"260-270"},"PeriodicalIF":3.4000,"publicationDate":"2025-03-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Analysis of spinal muscular atrophy patients from the spinal muscular atrophy and muscular dystrophy registry of Pakistan.\",\"authors\":\"Bisma Aziz, Ahmed A Arif, Kulsum Kazi, Salman Kirmani, Zeeshan Ansar, Asghar Nasir, Shahnaz Hamid Ibrahim, Khairunnisa Mukhtiar Ahmed, Zahra Hasan, Sara Khan\",\"doi\":\"10.1177/22143602241301657\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>BackgroundSpinal Muscular Atrophy (SMA) leads to motor neuron loss, with progressive muscle weakness and wasting. Nationwide registries for neuromuscular diseases are pivotal for assessing epidemiology, preparing for clinical trials, and for adopting standardized management guidelines.ObjectivesThis paper aims to present data gathered during the establishment of Pakistan's inaugural registry for genetically confirmed SMA cases.MethodsIn this retrospective study, 215 participants with genetically confirmed SMA were recruited. Telephonic interviews were conducted to collect data for the Muscular Disease Registry of Pakistan that was analyzed using STATA version 17.0.ResultsSMA type 1 was the most common type (71.2%, n = 153). Amongst patients who were tested for survival motor neuron (<i>SMN2</i>) copies, the majority (84.4%, n = 168) had two <i>SMN2</i> copies. SMA types were significantly associated with the ability to sit (p < 0.001) and walk (p < 0.001), and usage of a wheelchair (p = 0.0054). <i>SMN2</i> copy numbers were significantly associated with the ability to sit (p = 0.020) and walk (p = 0.031).ConclusionsThis study highlights the high prevalence of SMA genotypes and phenotypes associated with severe disease in our population. Our findings reiterate the challenging prognosis for Pakistani children with SMA and underscore the necessity of the development of nationwide newborn screening programs and making treatments available.</p>\",\"PeriodicalId\":16536,\"journal\":{\"name\":\"Journal of neuromuscular diseases\",\"volume\":\" \",\"pages\":\"260-270\"},\"PeriodicalIF\":3.4000,\"publicationDate\":\"2025-03-01\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Journal of neuromuscular diseases\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.1177/22143602241301657\",\"RegionNum\":4,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2024/12/20 0:00:00\",\"PubModel\":\"Epub\",\"JCR\":\"Q2\",\"JCRName\":\"CLINICAL NEUROLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Journal of neuromuscular diseases","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1177/22143602241301657","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2024/12/20 0:00:00","PubModel":"Epub","JCR":"Q2","JCRName":"CLINICAL NEUROLOGY","Score":null,"Total":0}
Analysis of spinal muscular atrophy patients from the spinal muscular atrophy and muscular dystrophy registry of Pakistan.
BackgroundSpinal Muscular Atrophy (SMA) leads to motor neuron loss, with progressive muscle weakness and wasting. Nationwide registries for neuromuscular diseases are pivotal for assessing epidemiology, preparing for clinical trials, and for adopting standardized management guidelines.ObjectivesThis paper aims to present data gathered during the establishment of Pakistan's inaugural registry for genetically confirmed SMA cases.MethodsIn this retrospective study, 215 participants with genetically confirmed SMA were recruited. Telephonic interviews were conducted to collect data for the Muscular Disease Registry of Pakistan that was analyzed using STATA version 17.0.ResultsSMA type 1 was the most common type (71.2%, n = 153). Amongst patients who were tested for survival motor neuron (SMN2) copies, the majority (84.4%, n = 168) had two SMN2 copies. SMA types were significantly associated with the ability to sit (p < 0.001) and walk (p < 0.001), and usage of a wheelchair (p = 0.0054). SMN2 copy numbers were significantly associated with the ability to sit (p = 0.020) and walk (p = 0.031).ConclusionsThis study highlights the high prevalence of SMA genotypes and phenotypes associated with severe disease in our population. Our findings reiterate the challenging prognosis for Pakistani children with SMA and underscore the necessity of the development of nationwide newborn screening programs and making treatments available.
期刊介绍:
The Journal of Neuromuscular Diseases aims to facilitate progress in understanding the molecular genetics/correlates, pathogenesis, pharmacology, diagnosis and treatment of acquired and genetic neuromuscular diseases (including muscular dystrophy, myasthenia gravis, spinal muscular atrophy, neuropathies, myopathies, myotonias and myositis). The journal publishes research reports, reviews, short communications, letters-to-the-editor, and will consider research that has negative findings. The journal is dedicated to providing an open forum for original research in basic science, translational and clinical research that will improve our fundamental understanding and lead to effective treatments of neuromuscular diseases.