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ASS1

argininosuccinate synthase 1

The ASS1 gene provides instructions for making argininosuccinate synthase 1, an enzyme vital for the urea cycle, which helps remove waste nitrogen from the body. The ASS1 gene encodes an enzyme called argininosuccinate synthase 1.

Chromosome 9q34.11 Autosomal recessive HGNC:758 Tier C
ASS1 9q34.11 p arm q arm 9

ASS1 is located on the long (q) arm of chromosome 9, at band 9q34.11. Arm ratio per GRCh38 - banding schematic.

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Overview

The ASS1 gene codes for the enzyme argininosuccinate synthase 1, which is a critical component of the urea cycle. This cycle is a series of biochemical reactions that primarily occur in liver cells, responsible for converting ammonia, a toxic waste product of protein metabolism, into urea, which can then be safely excreted from the body in urine. Maintaining a functional urea cycle is essential for preventing the accumulation of harmful nitrogenous compounds.

Variations within the ASS1 gene can affect the enzyme's function, potentially disrupting the urea cycle. Such disruptions can lead to the build-up of ammonia and other related compounds in the bloodstream, which can have significant health consequences.

What the gene does

The argininosuccinate synthase 1 enzyme, produced from the ASS1 gene, is instrumental in the third step of the urea cycle. This metabolic pathway is essential for the detoxification and removal of excess nitrogen generated from protein catabolism. Specifically, argininosuccinate synthase 1 catalyses the condensation reaction between citrulline and aspartate.

This reaction produces argininosuccinic acid, a crucial intermediate molecule in the urea cycle. Subsequent steps further process argininosuccinic acid to eventually form urea. By facilitating this key reaction, the ASS1 enzyme ensures the efficient conversion of ammonia into urea, preventing its accumulation to toxic levels in the body.

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Chromosome location

The ASS1 gene is located on chromosome 9, specifically at position 9q34.11. This means it is found on the long arm (q) of chromosome 9, within region 34, band 11. The precise location on the chromosome helps in mapping and understanding the genetic context of the gene.

Protein structure

The argininosuccinate synthase 1 protein consists of 412 amino acids. Domain architecture has not been experimentally characterised in detail for this protein.

Key variants

Variants within the ASS1 gene can alter the production or function of the argininosuccinate synthase 1 enzyme. These genetic changes can range from single nucleotide substitutions to larger deletions or insertions, and their impact depends on how they affect the enzyme's structure and activity. Pathogenic variants typically lead to a reduction or complete loss of enzyme function, impairing the urea cycle.

The table below shows the top 10 pathogenic or likely-pathogenic variants currently classified in ClinVar for ASS1.
View all on ClinVar →

Sample of pathogenic variants

10 pathogenic / likely-pathogenic variants from ClinVar, ranked by review status (expert-panel-reviewed first). This is a sample; recurrent founder variants in a specific population may not appear here - see the full ClinVar listing via the link above.

Variant (HGVS) Protein change Classification Evidence Associated condition
c.102T>G
single nucleotide variant
p.Tyr34Ter Pathogenic ★★☆☆ Citrullinemia type I
c.1168G>C
single nucleotide variant
p.Gly390Arg Pathogenic ★★☆☆ Citrullinemia
c.271A>C
single nucleotide variant
p.Thr91Pro Pathogenic ★★☆☆ Citrullinemia
c.364-2A>C
single nucleotide variant
- Pathogenic/Likely pathogenic ★★☆☆ Citrullinemia type I
c.537G>A
single nucleotide variant
p.Trp179Ter Pathogenic/Likely pathogenic ★★☆☆ Citrullinemia type I
c.571G>C
single nucleotide variant
p.Glu191Gln Pathogenic/Likely pathogenic ★★☆☆ Citrullinemia
c.631C>T
single nucleotide variant
p.Gln211Ter Pathogenic/Likely pathogenic ★★☆☆ Citrullinemia
c.773+1G>T
single nucleotide variant
- Pathogenic/Likely pathogenic ★★☆☆ Citrullinemia
c.815G>A
single nucleotide variant
p.Arg272His Pathogenic ★★☆☆ Citrullinemia type I
c.848del
Deletion
p.Glu283fs Pathogenic ★★☆☆ Citrullinemia type I

Evidence stars indicate ClinVar review status. Individual variant interpretation should always be performed by a qualified clinical laboratory - many variants remain classified as Variants of Uncertain Significance (VUS) pending more research.

Associated conditions

Pathogenic variants in the ASS1 gene are known to cause Citrullinaemia type I. This is an autosomal recessive inherited disorder, meaning an individual must inherit two copies of a pathogenic variant (one from each parent) to develop the condition. Citrullinaemia type I is characterised by the impaired function of the argininosuccinate synthase 1 enzyme, leading to an accumulation of ammonia and citrulline in the blood.

Inheritance pattern

Conditions caused by pathogenic ASS1 variants typically follow autosomal recessive inheritance.

Carrier parent 1 altered copy Carrier parent 1 altered copy Affected Carrier Carrier Unaffected Affected Carrier Unaffected Circles = females · Squares = males

When both parents are carriers, each child has a 25% chance of being affected, 50% of being a carrier, and 25% of being unaffected.

Carrier frequency by population How common is heterozygous ASS1 carrier status across ancestry groups?

UK clinical status

The ASS1 gene is recognised within several NHS Genomic Medicine Service national panels. It is listed with a 'green' status on panels including DDG2P, Foetal anomalies (R21), Hyperammonaemia, Intellectual disability, Likely inborn error of metabolism (R98), and Undiagnosed metabolic disorders. This indicates that there is strong evidence for the gene's association with these conditions and it is routinely considered in diagnostic testing.

Frequently asked questions

What is the urea cycle?

The urea cycle is a series of biochemical reactions that occur primarily in the liver. Its main function is to convert toxic ammonia, a waste product of protein breakdown, into urea, which is then safely excreted from the body through urine.

How does the ASS1 gene relate to Citrullinaemia type I?

Pathogenic variants in the ASS1 gene lead to a deficiency in the argininosuccinate synthase 1 enzyme. This enzyme is crucial for a step in the urea cycle, and its impaired function causes the build-up of ammonia and citrulline, which characterises Citrullinaemia type I.

Is Citrullinaemia type I an inherited condition?

Yes, Citrullinaemia type I is an autosomal recessive inherited disorder. This means that a child must inherit two copies of a pathogenic variant in the ASS1 gene, one from each parent, to be affected by the condition.

References

  1. Diez-Fernandez C, Rüfenacht V, Häberle J. Mutations in the Human Argininosuccinate Synthetase (ASS1) Gene, Impact on Patients, Common Changes, and Structural Considerations. Human mutation. 2017. PMID: 28111830
  2. Kose E, Unal O, Bulbul S. Identification of three novel mutations in fourteen patients with citrullinemia type 1. Clinical biochemistry. 2017. PMID: 28132756
  3. Husson A, Brasse-Lagnel C, Fairand A. Argininosuccinate synthetase from the urea cycle to the citrulline-NO cycle. European journal of biochemistry. 2003. PMID: 12709047
  4. Gao HZ, Kobayashi K, Tabata A. Identification of 16 novel mutations in the argininosuccinate synthetase gene and genotype-phenotype correlation in 38 classical citrullinemia patients. Human mutation. 2003. PMID: 12815590
  5. Häberle J, Pauli S, Linnebank M. Structure of the human argininosuccinate synthetase gene and an improved system for molecular diagnostics in patients with classical and mild citrullinemia. Human genetics. 2002. PMID: 11941481
  6. Adam MP, Bick S, Mirzaa GM. Urea Cycle Disorders Overview. 1993. PMID: 20301396
  7. Adam MP, Bick S, Mirzaa GM. Citrullinemia Type I. 1993. PMID: 20301631
Educational content. This page is not medical or genetic advice, is not individually reviewed by a clinician for each reader, and should not replace a consultation with a qualified healthcare professional or genetic counsellor. If you are considering genetic testing or acting on a test result, book a consultation.
Data sources Last updated 6 September 2026. Content compiled from HGNC · MedlinePlus Genetics · ClinGen · Genomics England PanelApp · NHS National Genomic Test Directory · ClinVar · UniProt · AlphaFold .