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Literature record

Case Report: Hyperprolinemia type II in a child with autism spectrum disorder and ALDH4A1 gene variant in a consanguineous family.

PMID 41602883 | PMCID PMC12832875 | DOI 10.3389/fped.2025.1726800 · Frontiers in pediatrics · 2025

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Hyperprolinemia type II (HPII) is a rare inherited metabolic disorder caused by the ALDH4A1 gene variant. Herein, we report a case of a preschool-aged Saudi girl who was born from consanguineous parents and presented with global developmental delay. The patient was clinically diagnosed with autism spectrum disorder with associated disruptive behaviors. Metabolic investigations revealed markedly elevated plasma and urinary proline levels, suggestive of a proline metabolism disorder. Whole-exome sequencing identified a homozygous variant of uncertain significance in the ALDH4A1 gene, which is associated with autosomal recessive HPII. Genetic testing of the patient's family members showed that all individuals had carrier status with varying zygosity. This case underscores the importance of metabolic and genetic evaluation in children with neurodevelopmental disorders and highlights that HPII can present with a clinical phenotype that overlaps substantially with ASD.

Validated evidence

TypeEntitySource evidenceConfidenceExtractor
geneALDH4A1“Case Report: Hyperprolinemia type II in a child with autism spectrum disorder and ALDH4A1 gene variant in a consanguineous family.”0.98hgnc_dict_v1
phenotypeautism spectrum disorder“Case Report: Hyperprolinemia type II in a child with autism spectrum disorder and ALDH4A1 gene variant in a consanguineous family.”0.98phenotype_alias_lexicon_v2
phenotypedevelopmental delay“Herein, we report a case of a preschool-aged Saudi girl who was born from consanguineous parents and presented with global developmental delay.”0.98phenotype_alias_lexicon_v2
phenotypeinborn error of metabolism“Hyperprolinemia type II (HPII) is a rare inherited metabolic disorder caused by the ALDH4A1 gene variant.”0.93phenotype_alias_lexicon_v2
phenotypeneurodevelopmental disorder“This case underscores the importance of metabolic and genetic evaluation in children with neurodevelopmental disorders and highlights that HPII can present with a clinical phenotype that overlaps substantially with ASD.”0.93phenotype_alias_lexicon_v2
populationSaudi Arabia“Case Report: Hyperprolinemia type II in a child with autism spectrum disorder and ALDH4A1 gene variant in a consanguineous family. Herein, we report a case of a preschool-aged Saudi girl who was born from consanguineous parents and presented with global developmental delay.”0.95saudi_context_rules_v1