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IEMbase 0103: SLC6A3-related dopamine transporter deficiency

Scope

Field Value
IEMbase ID 103
Nosology 23.1.05.01
Gene SLC6A3
External IDs OMIM:613135; OMIM:126455
Generated mapping MAPPED
Candidate DisMech targets Infantile_Parkinsonism-Dystonia.yaml
Review date 2026-07-07

IEMbase phenotype signal

IEMbase represents this as SLC6A3-related dopamine transporter deficiency, with alternate labels infantile parkinsonism-dystonia and DAT. Treatability is marked unknown and the cached JSON has no treatment rows.

The characteristic biochemical rows are increased CSF HVA/5-HIAA ratio, increased CSF homovanillic acid, and increased urinary homovanillic acid. The clinical rows are bulbar dysfunction, dyskinesia, dystonia, ocular flutter, and parkinsonism with hypokinetic features.

DisMech phenotype coverage

The generated mapping to Infantile_Parkinsonism-Dystonia.yaml is correct. The local entry describes dopamine transporter deficiency syndrome caused by biallelic SLC6A3 loss of function, impaired dopamine reuptake, dysregulated synaptic dopamine homeostasis, raised CSF HVA:5-HIAA ratio, and progressive nigrostriatal dysfunction.

Phenotype coverage includes parkinsonism-dystonia, dystonia, bradykinesia, rigidity, tremor, early hyperkinetic movement disorder, oculogyric crisis, axial hypotonia, delayed motor development, feeding difficulties, irritability, and decreased facial expression. Treatment coverage is broader than IEMbase: supportive care, tetrabenazine, benzodiazepines, dopamine agonists, physical therapy, and a note that levodopa is generally ineffective.

Concordance and completeness

Judgement: correct high-confidence mapping with high concordance.

IEMbase is more compact and emphasizes the diagnostic HVA/HVA:5-HIAA pattern and the bulbar/ocular-flutter rows. DisMech is richer for disease mechanism, progressive motor phenotype, treatment nuance, and the reason dopaminergic replacement is not analogous to synthesis-defect disorders.

Curation actions

  • Keep Infantile_Parkinsonism-Dystonia.yaml as the canonical target.
  • Consider adding urinary HVA and bulbar dysfunction or ocular flutter as review targets if the SLC6A3 entry is expanded.
  • No mapping correction needed.