Disease Directory Holt-Oram Syndrome
Connective Tissue

Holt-Oram Syndrome

Also known as: heart-hand syndrome, atriodigital dysplasia, TBX5 mutation

Prevalence

1 in 100,000

Onset

Congenital

Type

Genetic — autosomal dominant

Gene

TBX5

About Holt-Oram Syndrome

Holt-Oram syndrome is a highly penetrant autosomal dominant condition caused by pathogenic variants in TBX5, a T-box transcription factor essential for upper limb and cardiac development, resulting in the combination of preaxial upper limb anomalies and congenital heart defects. Upper limb malformations range from subtle abnormalities of the carpal or thumb bones detectable only radiographically to phocomelia, and are invariably present; cardiac defects, most commonly atrial septal defect and ventricular septal defect, occur in approximately 75% of individuals. Conduction system abnormalities, including atrioventricular block, occur even in the absence of structural heart defects and may present in adulthood.

Common Clinical Features

Preaxial upper limb abnormalities (triphalangeal, hypoplastic, or absent thumbs) Abnormal carpal or radial bone morphology on hand radiography Atrial septal defect (ostium secundum most common) Ventricular septal defect Atrioventricular conduction delay or complete heart block Unilateral or bilateral upper limb involvement (left side more severe) Phocomelia in severe cases

Clinical Trial Eligibility Tips

What to know before applying to Holt-Oram Syndrome trials.

Hand and wrist radiographs are required to document upper limb skeletal anomalies even if clinically subtle — abnormal carpal ossification pattern is a consistent finding used to confirm diagnosis.

Cardiac evaluation including ECG (for conduction abnormalities) and echocardiogram must be current; Holter monitoring may be requested if palpitations or presyncope are reported.

TBX5 molecular confirmation is required for enrolment in most trials; de novo variants are common, so family history may be negative and should not preclude genetic testing.

Patient Resources

Patient Organization

NORD (National Organization for Rare Disorders)

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Orphanet

European reference resource for rare diseases (ORPHA:392)

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NORD

National Organization for Rare Disorders

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Find recruiting Holt-Oram Syndrome trials

Search 500,000+ studies from ClinicalTrials.gov, filtered for Holt-Oram Syndrome. Updated daily.

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