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GeneE
26 sources retrieved Β· Most recent: April 2026 Β· Index updated 14 days ago
β“˜GeneE is for informational purposes only. It is not a substitute for professional medical advice, diagnosis, or treatment.
PAH
phenylalanine hydroxylase
Chromosome 12 Β· 12q23.2
NCBI Gene: 5053Ensembl: ENSG00000171759.11HGNC: HGNC:8582UniProt: P00439
323PubMed Papers
21Diseases
2Drugs
893Pathogenic Variants
RESEARCH IMPACT
TrendingVariant-Rich
CLINICAL
FDA Approved TargetOMIM Disease Gene
DATA QUALITY
βœ“ Experimental GO Evidenceβœ“ Swiss-Prot Reviewed
phenylalanine 4-monooxygenase activityL-phenylalanine catabolic processL-tyrosine biosynthetic processamino acid biosynthetic processphenylketonuriaHyperphenylalaninemiaMaternal hyperphenylalaninemiagenetic disorder
✦AI Summary

PAH (phenylalanine hydroxylase) is a cytosolic enzyme that catalyzes the hydroxylation of L-phenylalanine to L-tyrosine, serving as a critical step in phenylalanine catabolism and tyrosine biosynthesis. This conversion is essential for amino acid metabolism and supports downstream biosynthetic pathways including catecholamine production. PAH deficiency causes phenylalanine hydroxylase deficiency, a genetic disorder characterized by impaired conversion of phenylalanine to tyrosine, leading to elevated plasma phenylalanine levels and potential neurodevelopmental consequences if untreated. Clinically, phenylalanine hydroxylase deficiency is managed through early detection via newborn screening and dietary restriction of phenylalanine intake, which prevents accumulation of this amino acid and its toxic metabolites. The discovery and implementation of screening programs for this metabolic disorder represents a major success in preventive medicine, as early intervention can eliminate or substantially reduce the neurological complications historically associated with untreated disease. PAH variants have also been identified in genetic association studies, though the functional consequences of specific variants in other disease contexts remain to be fully elucidated.

Sources cited
1
This PMID discusses polycyclic aromatic hydrocarbons (PAHs as environmental compounds), not the phenylalanine hydroxylase gene
PMID: 35151703
2
This PMID discusses pulmonary arterial hypertension (PAH as a disease), not the phenylalanine hydroxylase gene
PMID: 31320454
3
This PMID discusses polycyclic aromatic hydrocarbons (PAHs as environmental compounds), not the phenylalanine hydroxylase gene
PMID: 36617662
4
This PMID discusses pulmonary arterial hypertension (PAH as a disease), not the phenylalanine hydroxylase gene
PMID: 33021809
5
This PMID discusses pulmonary arterial hypertension (PAH as a disease), not the phenylalanine hydroxylase gene
PMID: 34078089
6
This PMID discusses HIV-associated pulmonary arterial hypertension (PAH as a disease), not the phenylalanine hydroxylase gene
PMID: 28902721
7
This PMID discusses pulmonary arterial hypertension (PAH as a disease), not the phenylalanine hydroxylase gene
PMID: 40560058
8
This PMID discusses pulmonary arterial hypertension (PAH as a disease), not the phenylalanine hydroxylase gene
PMID: 37108634
Disease Associationsβ“˜21
phenylketonuriaOpen Targets
0.89Strong
HyperphenylalaninemiaOpen Targets
0.67Moderate
Maternal hyperphenylalaninemiaOpen Targets
0.66Moderate
genetic disorderOpen Targets
0.55Moderate
6-pyruvoyl-tetrahydropterin synthase deficiencyOpen Targets
0.53Moderate
BH4-deficient hyperphenylalaninemia AOpen Targets
0.53Moderate
pulmonary hypertension, primary, 1Open Targets
0.46Moderate
polymicrogyria, perisylvian, with cerebellar hypoplasia and arthrogryposisOpen Targets
0.42Moderate
malnutritionOpen Targets
0.37Weak
tetrahydrobiopterin-responsive hyperphenylalaninemia/phenylketonuriaOpen Targets
0.37Weak
breast cancerOpen Targets
0.34Weak
Combined pituitary hormone deficiencies, genetic formsOpen Targets
0.34Weak
mild hyperphenylalaninemiaOpen Targets
0.34Weak
Graves diseaseOpen Targets
0.33Weak
cervical carcinomaOpen Targets
0.33Weak
gastric cancerOpen Targets
0.32Weak
neurodegenerative diseaseOpen Targets
0.32Weak
bone remodeling diseaseOpen Targets
0.30Weak
propionic acidemiaOpen Targets
0.27Weak
rasopathyOpen Targets
0.27Weak
Phenylalanine hydroxylase deficiencyUniProt
Pathogenic Variants893
NM_000277.3(PAH):c.512G>A (p.Gly171Glu)Likely pathogenic
Phenylketonuria
β˜…β˜…β˜…β˜†2025β†’ Residue 171
NM_000277.3(PAH):c.716G>C (p.Gly239Ala)Pathogenic
not provided|Phenylketonuria
β˜…β˜…β˜…β˜†2025β†’ Residue 239
NM_000277.3(PAH):c.769G>A (p.Gly257Ser)Likely pathogenic
not provided|Phenylketonuria
β˜…β˜…β˜…β˜†2025β†’ Residue 257
NM_000277.3(PAH):c.547G>C (p.Glu183Gln)Likely pathogenic
not provided|Phenylketonuria
β˜…β˜…β˜…β˜†2025β†’ Residue 183
NM_000277.3(PAH):c.1054G>T (p.Gly352Cys)Pathogenic
not provided|Phenylketonuria
β˜…β˜…β˜…β˜†2025β†’ Residue 352
NM_000277.3(PAH):c.521T>A (p.Ile174Asn)Likely pathogenic
Phenylketonuria
β˜…β˜…β˜…β˜†2025β†’ Residue 174
NM_000277.3(PAH):c.361T>C (p.Phe121Leu)Pathogenic
Phenylketonuria
β˜…β˜…β˜…β˜†2025β†’ Residue 121
NM_000277.3(PAH):c.715G>A (p.Gly239Ser)Pathogenic
not provided|Phenylketonuria
β˜…β˜…β˜…β˜†2025β†’ Residue 239
NM_000277.3(PAH):c.183C>A (p.Asn61Lys)Likely pathogenic
Phenylketonuria
β˜…β˜…β˜…β˜†2025β†’ Residue 61
NM_000277.3(PAH):c.470G>A (p.Arg157Lys)Likely pathogenic
not provided|Phenylketonuria
β˜…β˜…β˜…β˜†2025β†’ Residue 157
NM_000277.3(PAH):c.911A>C (p.Gln304Pro)Likely pathogenic
Phenylketonuria
β˜…β˜…β˜…β˜†2025β†’ Residue 304
NM_000277.3(PAH):c.929C>A (p.Ser310Tyr)Pathogenic
not provided|Phenylketonuria
β˜…β˜…β˜…β˜†2025β†’ Residue 310
NM_000277.3(PAH):c.470G>T (p.Arg157Ile)Likely pathogenic
not provided|Phenylketonuria
β˜…β˜…β˜…β˜†2025β†’ Residue 157
NM_000277.3(PAH):c.716G>A (p.Gly239Asp)Pathogenic
not provided|Phenylketonuria
β˜…β˜…β˜…β˜†2025β†’ Residue 239
NM_000277.3(PAH):c.1229T>C (p.Phe410Ser)Pathogenic
not provided|Phenylketonuria
β˜…β˜…β˜…β˜†2025β†’ Residue 410
NM_000277.3(PAH):c.1183G>C (p.Ala395Pro)Pathogenic
not provided|Phenylketonuria
β˜…β˜…β˜…β˜†2025β†’ Residue 395
NM_000277.3(PAH):c.187_190delinsCCCA (p.Thr63_His64delinsProAsn)Likely pathogenic
Phenylketonuria|not provided
β˜…β˜…β˜…β˜†2025β†’ Residue 63
NM_000277.3(PAH):c.181A>G (p.Asn61Asp)Likely pathogenic
not provided|Phenylketonuria
β˜…β˜…β˜…β˜†2025β†’ Residue 61
NM_000277.3(PAH):c.1066-11G>APathogenic
Phenylketonuria|not provided|Inborn genetic diseases|Hyperphenylalaninemia
β˜…β˜…β˜…β˜†2024
NM_000277.3(PAH):c.452A>G (p.Asp151Gly)Likely pathogenic
not provided|Phenylketonuria
β˜…β˜…β˜…β˜†2024β†’ Residue 151
View on ClinVar β†—
Drug Targets2
SAPROPTERINApproved
Phenylalanine-4-hydroxylase activator
malnutrition
SAPROPTERIN DIHYDROCHLORIDEApproved
Phenylalanine-4-hydroxylase activator
phenylketonuria
Related Genes
QDPRProtein interaction99%DHFR2Protein interaction99%PTSProtein interaction99%AKR1B1Protein interaction98%DHFRProtein interaction98%SPRProtein interaction98%
Tissue Expression6 tissues
Liver
100%
Brain
0%
Ovary
0%
Heart
0%
Lung
0%
Bone Marrow
0%
Gene Interaction Network
Click a node to explore
PAHQDPRDHFR2PTSAKR1B1DHFRSPR
PROTEIN STRUCTURE
Preparing viewer…
PDB1J8U Β· 1.50 Γ… Β· X-ray
View on RCSB β†—
Constraintβ“˜
LOEUFβ“˜
1.26LoF Tolerant
pLIβ“˜
0.00Tolerant
Observed/Expected LoF1.01 [0.82–1.26]
RankingsWhere PAH stands among ~20K protein-coding genes
  • #1,028of 20,598
    Most Researched323 Β· top 5%
  • #531of 1,025
    FDA-Approved Drug Targets2
  • #46of 5,498
    Most Pathogenic Variants893 Β· top 1%
  • #13,336of 17,882
    Most Constrained (LOEUF)1.26
Genes detectedPAH
Sources retrieved26 papers
Response timeβ€”
πŸ“„ Sources
26β–Ό
1
Polycyclic aromatic hydrocarbon and its effects on human health: An overeview.
PMID: 35151703
Chemosphere Β· 2022
1.00
2
CCR2/CCR5-mediated macrophage-smooth muscle cell crosstalk in pulmonary hypertension.
PMID: 31320454
Eur Respir J Β· 2019
0.90
3
Polycyclic aromatic hydrocarbons (PAHs): Updated aspects of their determination, kinetics in the human body, and toxicity.
PMID: 36617662
J Toxicol Environ Health B Crit Rev Β· 2023
0.80
4
Single-Cell Study of Two Rat Models of Pulmonary Arterial Hypertension Reveals Connections to Human Pathobiology and Drug Repositioning.
PMID: 33021809
Am J Respir Crit Care Med Β· 2021
0.70
5
Stress Granule Assembly in Pulmonary Arterial Hypertension.
PMID: 39513903
Cells Β· 2024
0.68