دورية أكاديمية
أعرض في EDS

The male-to-female ratio in late-onset multiple acyl-CoA dehydrogenase deficiency: a systematic review and meta-analysis.

التفاصيل البيبلوغرافية
العنوان: The male-to-female ratio in late-onset multiple acyl-CoA dehydrogenase deficiency: a systematic review and meta-analysis.
المؤلفون: Ma J; Department of Neurology, First Hospital, Shanxi Medical University, No.85, Jiefang South Street, Taiyuan, China.; First Clinical Medical College, Shanxi Medical University, Taiyuan, China., Zhang H; Department of Neurology, First Hospital, Shanxi Medical University, No.85, Jiefang South Street, Taiyuan, China.; First Clinical Medical College, Shanxi Medical University, Taiyuan, China., Liang F; Department of Neurology, First Hospital, Shanxi Medical University, No.85, Jiefang South Street, Taiyuan, China.; First Clinical Medical College, Shanxi Medical University, Taiyuan, China., Li G; Department of Neurology, First Hospital, Shanxi Medical University, No.85, Jiefang South Street, Taiyuan, China.; First Clinical Medical College, Shanxi Medical University, Taiyuan, China., Pang X; Department of Neurology, First Hospital, Shanxi Medical University, No.85, Jiefang South Street, Taiyuan, China., Zhao R; Department of Neurology, First Hospital, Shanxi Medical University, No.85, Jiefang South Street, Taiyuan, China., Wang J; Department of Neurology, First Hospital, Shanxi Medical University, No.85, Jiefang South Street, Taiyuan, China., Chang X; Department of Neurology, First Hospital, Shanxi Medical University, No.85, Jiefang South Street, Taiyuan, China., Guo J; Department of Neurology, First Hospital, Shanxi Medical University, No.85, Jiefang South Street, Taiyuan, China. neuroguo@163.com., Zhang W; Department of Neurology, First Hospital, Shanxi Medical University, No.85, Jiefang South Street, Taiyuan, China. zhangvey@126.com.
المصدر: Orphanet journal of rare diseases [Orphanet J Rare Dis] 2024 Feb 16; Vol. 19 (1), pp. 72. Date of Electronic Publication: 2024 Feb 16.
نوع المنشور: Meta-Analysis; Systematic Review; Journal Article
اللغة: English
بيانات الدورية: Publisher: BioMed Central Country of Publication: England NLM ID: 101266602 Publication Model: Electronic Cited Medium: Internet ISSN: 1750-1172 (Electronic) Linking ISSN: 17501172 NLM ISO Abbreviation: Orphanet J Rare Dis Subsets: MEDLINE
أسماء مطبوعة: Original Publication: [London] : BioMed Central, 2006-
مواضيع طبية MeSH: Multiple Acyl Coenzyme A Dehydrogenase Deficiency*/genetics , Multiple Acyl Coenzyme A Dehydrogenase Deficiency*/diagnosis , Multiple Acyl Coenzyme A Dehydrogenase Deficiency*/metabolism , Iron-Sulfur Proteins*/genetics , Iron-Sulfur Proteins*/metabolism , Oxidoreductases Acting on CH-NH Group Donors*/genetics , Oxidoreductases Acting on CH-NH Group Donors*/metabolism, Humans ; Male ; Female ; Mutation ; Delayed Diagnosis ; Electron-Transferring Flavoproteins/genetics ; Acyl-CoA Dehydrogenase/genetics ; Acyl-CoA Dehydrogenase/metabolism
مستخلص: Background: Late-onset multiple acyl-CoA dehydrogenase deficiency (MADD) is the most common lipid storage myopathy. There are sex differences in fat metabolism and it is not known whether late-onset MADD affects men and women equally.
Methods: In this systematic review and meta-analysis, the PubMed, Embase, Web of Science, CNKI, CBM, and Wanfang databases were searched until 01/08/2023. Studies reporting sex distribution in patients with late-onset MADD were included. Two authors independently screened studies for eligibility, extracted data, and assessed risk of bias. Pre-specified outcomes of interest were the male-to-female ratio (MFR) of patients with late-onset MADD, the differences of clinical characteristics between the sexes, and factors influencing the MFR.
Results: Of 3379 identified studies, 34 met inclusion criteria, yielding a total of 609 late-onset MADD patients. The overall pooled percentage of males was 58% (95% CI, 54-63%) with low heterogeneity across studies (I 2  = 2.99%; P = 0.42). The mean onset ages, diagnostic delay, serum creatine kinase (CK), and allelic frequencies of 3 hotspot variants in ETFDH gene were similar between male and female patients (P > 0.05). Meta-regressions revealed that ethnic group was associated with the MFR in late-onset MADD, and subgroup meta-analyses demonstrated that East-Asian patients had a higher percentage of male, lower CK, and higher proportion of hotspot variants in ETFDH gene than non-East-Asian patients (P < 0.05).
Conclusions: Male patients with late-onset MADD were more common than female patients. Ethnicity was proved to be a factor influencing the MFR in late-onset MADD. These findings suggest that male sex may be a risk factor for the disease.
(© 2024. The Author(s).)
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معلومات مُعتمدة: 20210302123245 Shanxi Science and Technology Department
فهرسة مساهمة: Keywords: Ethnicity; Late-onset multiple acyl-CoA dehydrogenase deficiency; Male-to-female ratio; Meta-analysis; Sex difference
المشرفين على المادة: 0 (Electron-Transferring Flavoproteins)
0 (Iron-Sulfur Proteins)
EC 1.5.- (Oxidoreductases Acting on CH-NH Group Donors)
EC 1.3.8.7 (Acyl-CoA Dehydrogenase)
تواريخ الأحداث: Date Created: 20240216 Date Completed: 20240219 Latest Revision: 20240219
رمز التحديث: 20240219
مُعرف محوري في PubMed: PMC10873946
DOI: 10.1186/s13023-024-03072-6
PMID: 38365830
قاعدة البيانات: MEDLINE
الوصف
تدمد:1750-1172
DOI:10.1186/s13023-024-03072-6