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GeneE
10 sources retrieved Β· Most recent: April 2026 Β· Index updated 14 days ago
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ARSL
arylsulfatase L
Chromosome X Β· Xp22.33
NCBI Gene: 415Ensembl: ENSG00000157399HGNC: HGNC:719UniProt: A0A5F9ZHX8
31PubMed Papers
21Diseases
0Drugs
41Pathogenic Variants
FUNCTIONAL ROLE
Highly Constrained
CLINICAL
OMIM Disease Gene
DATA QUALITY
βœ“ Experimental GO Evidenceβœ“ Swiss-Prot Reviewed
Golgi apparatusarylsulfatase activityextracellular exosomeskeletal system developmentX-linked chondrodysplasia punctata 1chondrodysplasia punctata, brachytelephalangic, autosomalgenetic disorderchondrodysplasia punctata
✦AI Summary

ARSL (arylsulfatase L) is an X-linked gene encoding an arylsulfatase enzyme that exhibits catalytic activity toward artificial sulfate substrates like 4-methylumbelliferyl sulfate 12, but lacks activity toward steroid sulfates 1. The enzyme is essential for correct composition of cartilage and bone matrix during skeletal development 1. ARSL localizes to the endoplasmic reticulum lumen and Golgi apparatus, functioning in extracellular contexts [GO annotations]. Loss-of-function variants in ARSL cause X-linked recessive chondrodysplasia punctata 1 (CDPX1), a rare congenital skeletal dysplasia characterized by stippled epiphyses, nasal hypoplasia, brachydactyly, and shortened long bones 34. Prenatal manifestations include bony stippling (41% of cases), nasal hypoplasia (55%), shortened long bones (23%), and intrauterine complications including fetal death and intraventricular hemorrhage 3. Genetic heterogeneity presents diagnostic challenges; prenatal diagnosis relies on whole exome sequencing and molecular confirmation 4. Notably, maternal X-chromosome X disomy can cause CDPX1 in females through homozygosity of pathogenic ARSL variants, representing an atypical inheritance mechanism 5. These findings underscore ARSL's critical role in skeletal development and establish it as the primary genetic determinant of CDPX1.

Sources cited
1
ARSL exhibits arylsulfatase activity toward 4-methylumbelliferyl sulfate, lacks activity toward steroid sulfates, and is essential for cartilage and bone matrix composition
PMID: 7720070
2
ARSL exhibits arylsulfatase activity toward 4-methylumbelliferyl sulfate
PMID: 9497243
3
ARSL variants cause CDPX1 with prenatal manifestations including nasal hypoplasia (55%), bony stippling (41%), shortened long bones (23%), and intrauterine complications
PMID: 39313411
4
Novel frameshift deletion variant in ARSL causes CDPX1 with fetal nasal hypoplasia and stippled epiphyses; prenatal diagnosis confirmed via whole exome sequencing
PMID: 39425194
5
Maternal X-chromosome uniparental disomy can cause CDPX1 in females through ARSL variant homozygosity, representing a rare inheritance mechanism for X-linked recessive conditions
PMID: 35256563
Disease Associationsβ“˜21
X-linked chondrodysplasia punctata 1Open Targets
0.80Strong
chondrodysplasia punctata, brachytelephalangic, autosomalOpen Targets
0.53Moderate
genetic disorderOpen Targets
0.41Moderate
chondrodysplasia punctataOpen Targets
0.38Weak
Coffin-Siris syndrome 1Open Targets
0.27Weak
connective tissue diseaseOpen Targets
0.17Weak
dystonia, early-onset, and/or spastic paraplegiaOpen Targets
0.12Weak
Barrett's esophagusOpen Targets
0.06Suggestive
esophageal adenocarcinomaOpen Targets
0.04Suggestive
neoplasmOpen Targets
0.02Suggestive
melanomaOpen Targets
0.02Suggestive
mucopolysaccharidosis type 6Open Targets
0.02Suggestive
metachromatic leukodystrophyOpen Targets
0.01Suggestive
non-small cell lung carcinomaOpen Targets
0.01Suggestive
X-linked chondrodysplasia punctataOpen Targets
0.01Suggestive
chronic obstructive pulmonary diseaseOpen Targets
0.01Suggestive
Down syndromeOpen Targets
0.01Suggestive
ataxia telangiectasiaOpen Targets
0.01Suggestive
infectionOpen Targets
0.01Suggestive
metabolic syndromeOpen Targets
0.01Suggestive
Chondrodysplasia punctata 1, X-linked recessiveUniProt
Pathogenic Variants41
NM_000047.3(ARSL):c.331C>T (p.Arg111Cys)Likely pathogenic
not provided
β˜…β˜…β˜†β˜†2025β†’ Residue 111
NM_000047.3(ARSL):c.24-2A>GLikely pathogenic
Chondrodysplasia punctata, brachytelephalangic, autosomal|not provided
β˜…β˜…β˜†β˜†2023
NM_000047.3(ARSL):c.349G>A (p.Gly117Arg)Pathogenic
X-linked chondrodysplasia punctata 1|not provided
β˜…β˜…β˜†β˜†2023β†’ Residue 117
NM_000047.3(ARSL):c.217G>A (p.Gly73Ser)Likely pathogenic
X-linked chondrodysplasia punctata 1|Chondrodysplasia punctata, brachytelephalangic, autosomal
β˜…β˜…β˜†β˜†2022β†’ Residue 73
NM_000047.3(ARSL):c.1289+1G>APathogenic
X-linked chondrodysplasia punctata 1|Nonpapillary renal cell carcinoma
β˜…β˜…β˜†β˜†2022
NM_000047.3(ARSL):c.258C>A (p.Cys86Ter)Likely pathogenic
X-linked chondrodysplasia punctata 1
β˜…β˜†β˜†β˜†2025β†’ Residue 86
NM_000047.3(ARSL):c.854G>A (p.Arg285Lys)Likely pathogenic
not provided
β˜…β˜†β˜†β˜†2025β†’ Residue 285
NM_000047.3(ARSL):c.1387G>A (p.Ala463Thr)Pathogenic
not provided
β˜…β˜†β˜†β˜†2025β†’ Residue 463
NM_000047.3(ARSL):c.119T>G (p.Ile40Ser)Likely pathogenic
X-linked chondrodysplasia punctata 1|not provided
β˜…β˜†β˜†β˜†2025β†’ Residue 40
NM_000047.3(ARSL):c.30T>A (p.Cys10Ter)Pathogenic
Chondrodysplasia punctata, brachytelephalangic, autosomal
β˜…β˜†β˜†β˜†2024β†’ Residue 10
NM_000047.3(ARSL):c.1742G>A (p.Trp581Ter)Pathogenic
not provided
β˜…β˜†β˜†β˜†2024β†’ Residue 581
NM_000047.3(ARSL):c.131_140del (p.Met44fs)Pathogenic
Chondrodysplasia punctata, brachytelephalangic, autosomal
β˜…β˜†β˜†β˜†2024β†’ Residue 44
NM_000047.3(ARSL):c.540T>A (p.Asp180Glu)Likely pathogenic
not provided
β˜…β˜†β˜†β˜†2024β†’ Residue 180
NM_000047.3(ARSL):c.23+1G>CLikely pathogenic
Chondrodysplasia punctata, brachytelephalangic, autosomal
β˜…β˜†β˜†β˜†2023
NM_000047.3(ARSL):c.991+2T>CLikely pathogenic
X-linked chondrodysplasia punctata 1
β˜…β˜†β˜†β˜†2023
NM_000047.3(ARSL):c.1226C>T (p.Thr409Met)Likely pathogenic
not provided
β˜…β˜†β˜†β˜†2023β†’ Residue 409
NM_000047.3(ARSL):c.916A>G (p.Thr306Ala)Pathogenic
X-linked chondrodysplasia punctata 1
β˜…β˜†β˜†β˜†2022β†’ Residue 306
NM_000047.3(ARSL):c.1618C>T (p.Arg540Ter)Pathogenic
Chondrodysplasia punctata, brachytelephalangic, autosomal
β˜…β˜†β˜†β˜†2022β†’ Residue 540
NM_000047.3(ARSL):c.74del (p.Leu25fs)Pathogenic
Chondrodysplasia punctata, brachytelephalangic, autosomal
β˜…β˜†β˜†β˜†2021β†’ Residue 25
NM_000047.3(ARSL):c.185+1G>ALikely pathogenic
Chondrodysplasia punctata, brachytelephalangic, autosomal
β˜…β˜†β˜†β˜†2021
View on ClinVar β†—
Related Genes
PAPSS1Shared pathway100%PRELPShared pathway100%TNFRSF11BShared pathway100%SHOXProtein interaction95%EBPProtein interaction84%XGProtein interaction75%
Tissue Expression6 tissues
Liver
100%
Ovary
8%
Bone Marrow
3%
Heart
2%
Brain
1%
Lung
1%
Gene Interaction Network
Click a node to explore
ARSLPAPSS1PRELPTNFRSF11BSHOXEBPXG
PROTEIN STRUCTURE
Preparing viewer…
AlphaFoldAI-predicted Β· UniProt P51690
View on AlphaFold β†—
Constraintβ“˜
LOEUFβ“˜
0.29Highly Constrained
pLIβ“˜
1.00Intolerant
Observed/Expected LoF0.13 [0.06–0.29]
RankingsWhere ARSL stands among ~20K protein-coding genes
  • #11,665of 20,598
    Most Researched31
  • #1,527of 5,498
    Most Pathogenic Variants41
  • #1,094of 17,882
    Most Constrained (LOEUF)0.29 Β· top 10%
Genes detectedARSL
Sources retrieved10 papers
Response timeβ€”
πŸ“„ Sources
10β–Ό
1
Prenatal Ultrasonographic Features Associated With ARSL and X-Linked Chondrodysplasia Punctata 1 (CDPX1): Literature Review and Case Series.
PMID: 39313411
Prenat Diagn Β· 2024
1.00
2
PMID: 20301713
0.90
3
A novel frameshift deletion variant of ARSL associated with X-linked recessive chondrodysplasia punctata 1: a case report and literature review of prenatal, confirmed cases.
PMID: 39425194
BMC Med Genomics Β· 2024
0.80
4
Preparation, Physicochemical Properties, and In Vitro Toxicity towards Cancer Cells of Novel Types of Arsonoliposomes.
PMID: 32268585
Pharmaceutics Β· 2020
0.70
5
Continuous Arabic Sign Language Recognition Models.
PMID: 40363353
Sensors (Basel) Β· 2025
0.60