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小儿1型戊二酸尿症:14例病例、诊断与管理

Pediatric Glutaric Aciduria Type 1: 14 Cases, Diagnosis and Management.

作者信息

Cornelius Leema P, Raju Vivekasaravanan, Julin Asir

机构信息

Department of Paediatric Neurology, Institute of Child Health and Hospital for Children, Madras Medical College, Chennai, Tamil Nadu, India.

出版信息

Ann Indian Acad Neurol. 2021 Jan-Feb;24(1):22-26. doi: 10.4103/aian.AIAN_42_20. Epub 2020 Dec 28.

Abstract

INTRODUCTION

Glutaric aciduria type I is an autosomal recessive disorder of lysine metabolism due to the defect of the enzyme glutaryl-CoA dehydrogenase. The regression of milestones following an intercurrent infection with disabling dystonia is the common presentation. We report the clinical features, diagnosis, and management of 14 south Indian children with glutaric aciduria type I.

RESULTS

Males predominated the study (57.1%). The mean age of onset of the symptoms was 8.57 ± 3.57 months. The mean age at the time of diagnosis was 35.21 ± 48.31 months. The history of consanguinity was noted in 57.1%. Development was normal prior to the onset of acute crises in nearly three fourths. Acute crises triggered by infection followed by the regression of milestones was the major presenting feature in 10 children (71.4%). Macrocephaly was another prominent feature in an equal number. Bat's wing appearance (fronto temporal atrophy) was present in all children. Nearly 80% had moderate to severe disability in the form of dystonic movement disorder and spastic quadriparesis.

CONCLUSION

Glutaric aciduria type Ihas to be identified and managed early to have a better outcome.

摘要

引言

I型戊二酸尿症是一种常染色体隐性赖氨酸代谢紊乱疾病,由戊二酰辅酶A脱氢酶缺陷引起。在并发感染后出现发育里程碑倒退并伴有致残性肌张力障碍是常见表现。我们报告了14例南印度I型戊二酸尿症儿童的临床特征、诊断及治疗情况。

结果

男性在研究中占主导(57.1%)。症状出现的平均年龄为8.57±3.57个月。诊断时的平均年龄为35.21±48.31个月。57.1%的患儿有近亲结婚史。近四分之三的患儿在急性发作前发育正常。10名患儿(71.4%)的主要表现为感染引发急性发作,随后发育里程碑倒退。同样数量的患儿有大头畸形这一突出特征。所有患儿均有蝙蝠翼样外观(额颞叶萎缩)。近80%的患儿有中度至重度残疾,表现为肌张力障碍性运动障碍和痉挛性四肢瘫。

结论

必须尽早识别并治疗I型戊二酸尿症,以获得更好的治疗效果。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5ed3/8061498/3db77c65c1e8/AIAN-24-22-g001.jpg

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