Qian Guling, Hong Fang, Tong Fan, et al. Clinical and Genotype Analysis of 16 Cases of Very Long Chain Acyl-CoA Dehydrogenase Deficiency. [J]. 26(9):8-11(2020)
DOI:
Qian Guling, Hong Fang, Tong Fan, et al. Clinical and Genotype Analysis of 16 Cases of Very Long Chain Acyl-CoA Dehydrogenase Deficiency. [J]. 26(9):8-11(2020) DOI: 10.13407/j.cnki.jpp.1672-108X.2020.09.003.
Clinical and Genotype Analysis of 16 Cases of Very Long Chain Acyl-CoA Dehydrogenase Deficiency
Objective: To probe into the clinical manifestation
laboratory examination
diagnosis and treatment outcome of very long-chain acyl-CoA dehydrogenase deficiency (VLCADD)
and the relationship between clinical phenotype and genotype. Methods: A total of 16 children with VLCADD after neonatal screening and clinical diagnosis in our hospital from Jun. 2013 to Jun. 2019 were extracted. Clinical manifestation
laboratory examination and genotype were retrospectively analyzed. Results: Among the 16 cases
there were 5 males and 11 females
aged from 7 months to 10 years. There were 3 cases (18.75%) of myocardial type
5 cases (31.25%) of liver type
7 cases (43.75%) of muscular type
and 1 case was lost to follow-up. There were 3 children with myocardial type died
2 children with liver type died
and there was no death for muscular type. A total of 28 different genotypes were detected in 16 children
and 12 of them were unreported new mutations. Among the 28 genotypes
there are 24 point mutations
3 small fragment deletions
and 1 single base insertion. Conclusion: VLCADD in severe children are start early and with critical onset
and it is difficult to reverse the outcome even if the baby was early diagnosed through neonatal screening. A variant with at least one null allele or a frame shift in the genotype may indicate a serious phenotype.