Orofaciodigital syndrome type 1

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ORPHA:2750OMIM:311200Q87.0
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3Active trials2Specialists8Treatment centers

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Overview

Orofaciodigital syndrome type 1 (OFD1), also known as Papillon-Léage-Psaume syndrome, is a rare genetic disorder that primarily affects the development of the oral cavity, face, and digits (fingers and toes). It is caused by pathogenic variants in the OFD1 gene (also known as CXORF5), located on the X chromosome. This condition is typically lethal in males during embryonic development, so it is observed almost exclusively in females. The hallmark features of OFD1 involve multiple body systems. Oral abnormalities include a lobed or bifid tongue, lingual hamartomas (benign tongue growths), cleft palate or high-arched palate, abnormal frenulae (bands of tissue connecting the lips and gums), and dental anomalies such as missing or extra teeth. Facial features may include facial asymmetry, milia (small skin cysts) particularly on the ears and face, median cleft lip, and a broad nasal root with hypoplasia of the nasal alae. Digital abnormalities include brachydactyly (short fingers or toes), syndactyly (fused digits), clinodactyly (curved digits), and polydactyly. Importantly, approximately 50% of affected individuals develop polycystic kidney disease, which can progress to renal failure and represents a major cause of morbidity and mortality. Central nervous system involvement, including agenesis of the corpus callosum, cerebellar abnormalities, and intellectual disability, occurs in a subset of patients. There is currently no cure for OFD1. Management is multidisciplinary and symptom-based. Surgical interventions may address cleft palate, tongue abnormalities, and polydactyly. Dental care is important for managing oral anomalies. Renal function should be monitored regularly due to the risk of polycystic kidney disease, and standard treatments for chronic kidney disease are applied as needed. Neurodevelopmental support, including speech therapy and educational interventions, may be beneficial for those with intellectual disability.

Also known as:

Clinical phenotype terms— hover any for plain English:

Lobulated tongueHP:0000180Broad alveolar ridgesHP:0000187Accessory oral frenulumHP:0000191Tongue nodulesHP:0000199
Inheritance

X-linked dominant

Carried on the X chromosome; a single copy can cause the condition

Age of Onset

Neonatal

Begins at or shortly after birth (first 4 weeks)

Orphanet ↗OMIM ↗NORD ↗

FDA & Trial Timeline

3 events
Oct 2025Optimal Strategy to Correct Stent underexpAnsion in Resistant Lesions

University Hospital, Clermont-Ferrand — NA

TrialRECRUITING
Apr 2016Pilot Study for TCE Imaging of the Esophagus Using an OFDI Capsule With a Compact Imaging System

Massachusetts General Hospital — NA

TrialRECRUITING
Jun 2014Surveillance and Image-Guided Biopsy of the Esophagus Using an OFDI Capsule

Massachusetts General Hospital — NA

TrialACTIVE NOT RECRUITING

Data sourced from FDA regulatory filings and ClinicalTrials.gov. Updated periodically.

Treatments

No FDA-approved treatments are currently listed for Orofaciodigital syndrome type 1.

3 clinical trialsare actively recruiting — trials can provide access to cutting-edge therapies.

View clinical trials →

Clinical Trials

3 recruitingView all trials with filters →
N/A3 trials
Pilot Study for TCE Imaging of the Esophagus Using an OFDI Capsule With a Compact Imaging System
N/A
Actively Recruiting
PI: Guillermo Tearney, MD, PhD (Massachusetts General Hospital) · Sites: Boston, Massachusetts · Age: 1485 yrs
Surveillance and Image-Guided Biopsy of the Esophagus Using an OFDI Capsule
N/A
Active
PI: Guillermo Tearney, MD, PhD (Massachusetts General Hospital) · Sites: Boston, Massachusetts · Age: 1899 yrs
Optimal Strategy to Correct Stent underexpAnsion in Resistant Lesions
N/A
Actively Recruiting
PI: Géraud Souteyrand, MD, PhD (CHU de Clermont-Ferrand) · Sites: Clermont-Ferrand, Auvergne; Clermont-Ferrand · Age: 1899 yrs

Specialists

2 foundView all specialists →
GS
Géraud Souteyrand
Specialist
PI on 1 active trial

Treatment Centers

8 centers
🏥 NORD

Baylor College of Medicine Rare Disease Center

Baylor College of Medicine

📍 Houston, TX

🏥 NORD

Stanford Medicine Rare Disease Center

Stanford Medicine

📍 Stanford, CA

🔬 UDN

NIH Clinical Center Undiagnosed Diseases Program

National Institutes of Health

📍 Bethesda, MD

🔬 UDN

UCLA UDN Clinical Site

UCLA Health

📍 Los Angeles, CA

🔬 UDN

Baylor College of Medicine UDN Clinical Site

Baylor College of Medicine

📍 Houston, TX

🔬 UDN

Harvard/MGH UDN Clinical Site

Massachusetts General Hospital

📍 Boston, MA

🏥 NORD

Mayo Clinic Center for Individualized Medicine

Mayo Clinic

📍 Rochester, MN

👤 Mayo Clinic Center for Individualized Medicine

🏥 NORD

UCLA Rare Disease Day Program

UCLA Health

📍 Los Angeles, CA

Travel Grants

No travel grants are currently matched to Orofaciodigital syndrome type 1.

Search all travel grants →NORD Financial Assistance ↗

Community

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Latest news about Orofaciodigital syndrome type 1

Disease timeline:

New recruiting trial: Optimal Strategy to Correct Stent underexpAnsion in Resistant Lesions

A new clinical trial is recruiting patients for Orofaciodigital syndrome type 1

New recruiting trial: Pilot Study for TCE Imaging of the Esophagus Using an OFDI Capsule With a Compact Imaging System

A new clinical trial is recruiting patients for Orofaciodigital syndrome type 1

Caregiver Resources

NORD Caregiver Resources

Support, advocacy, and financial assistance for caregivers of rare disease patients.

Mental Health Support

Rare disease caregiving can be isolating. Connect with counseling and peer support.

Family & Caregiver Grants

Financial assistance programs specifically for caregivers of rare disease patients.

Social Security Disability

Learn how rare disease patients may qualify for SSDI/SSI benefits.

Common questions about Orofaciodigital syndrome type 1

What is Orofaciodigital syndrome type 1?

Orofaciodigital syndrome type 1 (OFD1), also known as Papillon-Léage-Psaume syndrome, is a rare genetic disorder that primarily affects the development of the oral cavity, face, and digits (fingers and toes). It is caused by pathogenic variants in the OFD1 gene (also known as CXORF5), located on the X chromosome. This condition is typically lethal in males during embryonic development, so it is observed almost exclusively in females. The hallmark features of OFD1 involve multiple body systems. Oral abnormalities include a lobed or bifid tongue, lingual hamartomas (benign tongue growths), clef

How is Orofaciodigital syndrome type 1 inherited?

Orofaciodigital syndrome type 1 follows a x-linked dominant inheritance pattern. Genetic counseling can help families understand recurrence risk and testing options.

At what age does Orofaciodigital syndrome type 1 typically begin?

Typical onset of Orofaciodigital syndrome type 1 is neonatal. Age of onset can vary across affected individuals.

Are there clinical trials for Orofaciodigital syndrome type 1?

Yes — 3 recruiting clinical trials are currently listed for Orofaciodigital syndrome type 1 on UniteRare. See the clinical trials section on this page for phase, sponsor, and site details sourced from ClinicalTrials.gov.

Which specialists treat Orofaciodigital syndrome type 1?

2 specialists and care centers treating Orofaciodigital syndrome type 1 are listed on UniteRare, sourced from ClinicalTrials.gov principal investigators, published research, and the NPPES NPI registry.