HomeAboutRankingsData Sources
Β© 2026 GeneE
🧬
GeneE
10 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.
SHOX
SHOX homeobox
Chromosome X|Y Β· X;Y
NCBI Gene: 6473Ensembl: ENSG00000185960.15HGNC: HGNC:10853UniProt: O15266
138PubMed Papers
23Diseases
0Drugs
58Pathogenic Variants
FUNCTIONAL ROLE
Transcription Factor
RESEARCH IMPACT
Variant-Rich
CLINICAL
OMIM Disease Gene
DATA QUALITY
βœ“ Experimental GO Evidenceβœ“ Swiss-Prot Reviewed
positive regulation of transcription by RNA polymerase IInucleoplasmDNA-binding transcription activator activity, RNA polymerase II-specificprotein bindingLeri-Weill dyschondrosteosisLanger mesomelic dysplasiaSHOX-related short statureLΓ©ri-Weill dyschondrosteosis
✦AI Summary

SHOX is a pseudoautosomal homeobox transcription factor located on the short arms of both X and Y chrX|Y that plays a fundamental role in skeletal growth and development 1. As a sequence-specific DNA-binding transcription factor, SHOX preferentially binds DNA elements with the sequence 5'-TAATNNNATTA-3' and directly activates NPPB transcription in osteogenic cells, with strongest expression observed in bone marrow fibroblasts 1. SHOX escapes X-inactivation, allowing dosage-dependent effects across sex chromosome X|Y 1. SHOX haploinsufficiency causes multiple skeletal disorders with significant clinical implications. Heterozygous SHOX mutations occur in approximately 50-90% of Leri-Weill dyschondrosteosis cases, 2-15% of idiopathic short stature, and 66% of Turner syndrome patients, making SHOX defects the most common monogenic cause of short stature 2. Homozygous mutations result in Langer mesomelic dysplasia 3. Importantly, SHOX regulatory element alterations, including whole gene duplications lacking complete flanking regulatory elements, can paradoxically produce haploinsufficiency phenotypes 4. Clinically, SHOX gene haploinsufficiency is an FDA-approved indication for recombinant growth hormone therapy in pediatric patients 5, representing an important therapeutic option for improving adult height in affected children 6.

Sources cited
1
SHOX is a pseudoautosomal homeobox gene expressed in bone marrow fibroblasts that escapes X-inactivation; heterozygous mutations cause short stature and dyschondrosteosis
PMID: 10549307
2
Heterozygous SHOX defects occur in 50-90% of Leri-Weill dyschondrosteosis and 2-15% of idiopathic short stature, making SHOX defects the most important monogenic cause of short stature
PMID: 21150837
3
SHOX mutations occur in ~1 in 1000 newborns; heterozygous mutations affect extremity development and are implicated in Madelung's deformity, Leri-Weill dyschondrosteosis (77%), Turner's syndrome (66%), and idiopathic short stature (3%)
PMID: 19724992
4
SHOX whole gene duplications containing variable amounts of regulatory elements can result in SHOX haploinsufficiency phenotypes and are overrepresented in Leri-Weill dyschondrosteosis and idiopathic short stature
PMID: 36927524
5
SHOX gene haploinsufficiency is an FDA-approved indication for pediatric growth hormone therapy
PMID: 35985710
6
Patients with SHOX deficiency can be treated with recombinant growth hormone to improve adult height
PMID: 39857834
7
The SHOX gene plays a crucial role in short stature and abnormal skeletal phenotype in patients with Turner syndrome
PMID: 37408200
Disease Associationsβ“˜23
Leri-Weill dyschondrosteosisOpen Targets
0.80Strong
Langer mesomelic dysplasiaOpen Targets
0.77Strong
SHOX-related short statureOpen Targets
0.72Strong
LΓ©ri-Weill dyschondrosteosisOpen Targets
0.70Moderate
neurodegenerative diseaseOpen Targets
0.33Weak
COVID-19Open Targets
0.29Weak
severe acute respiratory syndromeOpen Targets
0.29Weak
femur fractureOpen Targets
0.11Weak
hyperinsulinemic hypoglycemia, familial, 4Open Targets
0.03Suggestive
clubfootOpen Targets
0.03Suggestive
Timothy syndromeOpen Targets
0.02Suggestive
Turner syndromeOpen Targets
0.02Suggestive
synovial sarcomaOpen Targets
0.02Suggestive
hypochondroplasiaOpen Targets
0.01Suggestive
Mayer-Rokitansky-Kuster-Hauser syndromeOpen Targets
0.01Suggestive
systemic lupus erythematosusOpen Targets
0.01Suggestive
scoliosisOpen Targets
0.01Suggestive
ovarian dysfunctionOpen Targets
0.01Suggestive
Premature ovarian insufficiencyOpen Targets
0.01Suggestive
chondrodysplasiaOpen Targets
0.01Suggestive
Langer mesomelic dysplasiaUniProt
Leri-Weill dyschondrosteosisUniProt
Short stature, idiopathic, X-linkedUniProt
Pathogenic Variants58
NM_000451.4(SHOX):c.517C>T (p.Arg173Cys)Pathogenic
Leri-Weill dyschondrosteosis|not provided|SHOX-related disorder
β˜…β˜…β˜†β˜†2025β†’ Residue 173
NM_000451.4(SHOX):c.518G>A (p.Arg173His)Pathogenic
not provided|Leri-Weill dyschondrosteosis
β˜…β˜…β˜†β˜†2025β†’ Residue 173
NM_000451.4(SHOX):c.502C>T (p.Arg168Trp)Pathogenic
Langer mesomelic dysplasia syndrome|Leri-Weill dyschondrosteosis|not provided
β˜…β˜…β˜†β˜†2025β†’ Residue 168
NM_000451.4(SHOX):c.352_353del (p.Arg118fs)Pathogenic
not provided|Leri-Weill dyschondrosteosis
β˜…β˜…β˜†β˜†2025β†’ Residue 118
NM_000451.4(SHOX):c.454C>T (p.Gln152Ter)Pathogenic
not provided|Leri-Weill dyschondrosteosis
β˜…β˜…β˜†β˜†2024β†’ Residue 152
NM_000451.4(SHOX):c.583C>T (p.Arg195Ter)Pathogenic
SHOX-related short stature|not provided|Leri-Weill dyschondrosteosis|SHOX-related disorder
β˜…β˜…β˜†β˜†2023β†’ Residue 195
NM_000451.4(SHOX):c.582C>A (p.Cys194Ter)Pathogenic
not provided
β˜…β˜…β˜†β˜†2023β†’ Residue 194
NM_000451.4(SHOX):c.-19G>APathogenic
SHOX-related short stature|Leri-Weill dyschondrosteosis
β˜…β˜…β˜†β˜†2022
NM_000451.4(SHOX):c.335A>C (p.Gln112Pro)Pathogenic
Leri-Weill dyschondrosteosis
β˜…β˜…β˜†β˜†2022β†’ Residue 112
NM_000451.4(SHOX):c.349C>T (p.Gln117Ter)Pathogenic
SHOX-related short stature|not provided
β˜…β˜…β˜†β˜†2020β†’ Residue 117
NM_000451.4(SHOX):c.400C>T (p.Arg134Ter)Pathogenic
not provided
β˜…β˜†β˜†β˜†2025β†’ Residue 134
NM_000451.4(SHOX):c.508G>C (p.Ala170Pro)Pathogenic
Langer mesomelic dysplasia syndrome|Leri-Weill dyschondrosteosis|not provided
β˜…β˜†β˜†β˜†2025β†’ Residue 170
NM_000451.4(SHOX):c.518G>T (p.Arg173Leu)Likely pathogenic
not provided
β˜…β˜†β˜†β˜†2025β†’ Residue 173
NM_000451.4(SHOX):c.421T>G (p.Tyr141Asp)Likely pathogenic
not provided
β˜…β˜†β˜†β˜†2025β†’ Residue 141
NM_000451.4(SHOX):c.458G>T (p.Arg153Leu)Likely pathogenic
Leri-Weill dyschondrosteosis
β˜…β˜†β˜†β˜†2025β†’ Residue 153
NM_000451.4(SHOX):c.379G>T (p.Glu127Ter)Likely pathogenic
Leri-Weill dyschondrosteosis
β˜…β˜†β˜†β˜†2025β†’ Residue 127
NM_000451.4(SHOX):c.278-1G>APathogenic
Short stature
β˜…β˜†β˜†β˜†2025
NM_000451.4(SHOX):c.503G>A (p.Arg168Gln)Likely pathogenic
Short stature|SHOX-related disorder
β˜…β˜†β˜†β˜†2024β†’ Residue 168
NM_000451.4(SHOX):c.425C>G (p.Pro142Arg)Likely pathogenic
Leri-Weill dyschondrosteosis
β˜…β˜†β˜†β˜†2024β†’ Residue 142
NM_000451.4(SHOX):c.675_676insA (p.Pro226fs)Likely pathogenic
Leri-Weill dyschondrosteosis
β˜…β˜†β˜†β˜†2024β†’ Residue 226
View on ClinVar β†—
Related Genes
TNFRSF11BShared pathway100%PRELPShared pathway100%PAPSS1Shared pathway100%ARSLProtein interaction95%CSF2RAProtein interaction78%STSProtein interaction76%
Tissue Expression6 tissues
Brain
100%
Ovary
84%
Liver
77%
Lung
71%
Bone Marrow
0%
Heart
0%
Gene Interaction Network
Click a node to explore
SHOXTNFRSF11BPRELPPAPSS1ARSLCSF2RASTS
PROTEIN STRUCTURE
Preparing viewer…
AlphaFoldAI-predicted Β· UniProt O15266
View on AlphaFold β†—
RankingsWhere SHOX stands among ~20K protein-coding genes
  • #3,352of 20,598
    Most Researched138 Β· top quartile
  • #1,196of 5,498
    Most Pathogenic Variants58 Β· top quartile
Genes detectedSHOX
Sources retrieved10 papers
Response timeβ€”
πŸ“„ Sources
10β–Ό
1
Clinical Indications for Growth Hormone Therapy.
PMID: 35985710
Adv Pediatr Β· 2022
1.00
2
Organ Abnormalities Caused by Turner Syndrome.
PMID: 37408200
Cells Β· 2023
0.90
3
PMID: 20301394
0.80
4
SHOX Whole Gene Duplications Are Overrepresented in SHOX Haploinsufficiency Phenotype Cohorts.
PMID: 36927524
Cytogenet Genome Res Β· 2022
0.70
5
GH Therapy in Non-Growth Hormone-Deficient Children.
PMID: 39857834
Children (Basel) Β· 2024
0.60