BBD Longitudinal Study of Osteogenesis Imperfecta

NCT ID: NCT02432625

Last Updated: 2025-08-13

Study Results

Results pending

The study team has not published outcome measurements, participant flow, or safety data for this trial yet. Check back later for updates.

Basic Information

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Recruitment Status

RECRUITING

Total Enrollment

1000 participants

Study Classification

OBSERVATIONAL

Study Start Date

2015-06-01

Study Completion Date

2031-12-31

Brief Summary

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Osteogenesis Imperfecta (OI) is a rare disorder of increased bone fragility characterized by fractures with minimal or absent trauma, dentinogenesis imperfecta (DI), and, in adult years, hearing loss. It is seen in both genders and all races. The clinical features of OI represent a continuum varying from perinatal lethality to individuals with severe skeletal deformities, mobility impairments, and very short stature to nearly asymptomatic individuals with a mild predisposition to fractures, normal stature, and normal lifespan. Fractures can occur in any bone, but are most common in the extremities. These disorders can be devastating and progressive and result in deformity, chronic pain, impaired function and loss of quality of life.

The overall goal of this study is to answer specific question about the natural history of brittle bone diseases as defined by molecular etiology and to develop the foundation for prospective clinical studies.

Detailed Description

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The purpose of this natural history study is to perform a long-term follow-up of a large group of people with osteogenesis imperfecta (OI). The research aims are:

1. To collect natural history data on all individuals enrolled in this longitudinal study. The cause of the brittle bone disease will be compared with things like severity, various features and response to treatments.
2. To determine how often people with type I OI have vertebral compression fractures of the spine.
3. To determine how often people with OI develop scoliosis (curvature of the spine).
4. To determine how often people with OI have problems with teeth alignment and how dental health impacts a person's quality of life.
5. To determine the effect of pregnancy in women with OI.

There will be a total of 1000 people with OI in this study. Participants will be asked to come in every year if 17Y and younger or every other year if 18Y and older for a total of five years.

The following information will be collected at the study visits:

Birth History and past surgical history, Current medical history, Scoliosis evaluation, Walking ability Questionnaire, Dental Quality of Life Questionnaire, Scoliosis and fractures Quality of Life Questionnaires, Physical development evaluation, Medications Use

The following tests will be performed:

Physical exam, dental exam, lung function test, hearing test, mobility test.

The following X-rays will be taken:

DEXA scan, X-ray of the spine, X-ray of the jaw.

Biospecimen (urine and blood) samples will be collected.

Conditions

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Osteogenesis Imperfecta

Study Design

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Observational Model Type

COHORT

Study Time Perspective

PROSPECTIVE

Eligibility Criteria

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Inclusion Criteria

* Individuals with OI diagnosed by molecular (DNA) analysis OR
* Individuals whose clinical history and radiographs are highly suggestive of OI, but whose diagnosis has not been verified by biochemical or molecular studies


• Patients with nonsense or frameshift mutations in COL1A1 or COL1A2 of any age and clinical features of OI type I.


* All study participants between the ages of 3 to 17 years OR
* Study participants 18 years and older with scoliosis

Dental and Craniofacial Abnormalities in OI component:


Pregnancy in OI component:


• Females of reproductive age with mutations in any known gene causing OI, who are contemplating pregnancy within 5 years of enrollment in the Natural History Study OR Females who are pregnant with available pre-pregnancy BMD (within 5 years prior to the first pregnancy visit).

Exclusion Criteria

* Individuals who are unable to return for their scheduled follow up visits.
* Individuals with skeletal dysplasias other than OI
* Individuals with OI and a second genetic or syndromic diagnosis


* Use of a bone-acting treatment agent such as bisphosphonates, calcitonin, calcitriol, fluoride, etc., within one year of enrollment.
* Conditions other than Osteogenesis Imperfecta-HaploInsufficiency (OI-HI) affecting muscle and/or bone development (i.e. cerebral palsy, rickets)
* Nonsense or frame shift mutations in the final coding exons of COL1A1 or COL1A2, as this may not lead to haploinsufficiency.

Scoliosis in OI component:


* Males
* Females who are peri-menopausal or menopausal
* Females who had gestations associated with higher order multiples.
Eligible Sex

ALL

Accepts Healthy Volunteers

No

Sponsors

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Shriners Hospitals for Children

OTHER

Sponsor Role collaborator

Hospital for Special Surgery, New York

OTHER

Sponsor Role collaborator

Children's National Research Institute

OTHER

Sponsor Role collaborator

Hugo W. Moser Research Institute at Kennedy Krieger, Inc.

OTHER

Sponsor Role collaborator

University of California, Los Angeles

OTHER

Sponsor Role collaborator

Oregon Health and Science University

OTHER

Sponsor Role collaborator

University of Nebraska

OTHER

Sponsor Role collaborator

Alfred I. duPont Hospital for Children

OTHER

Sponsor Role collaborator

University of South Florida

OTHER

Sponsor Role collaborator

Phoenix Children's Hospital

OTHER

Sponsor Role collaborator

Marquette University

OTHER

Sponsor Role collaborator

Baylor College of Medicine

OTHER

Sponsor Role lead

Responsible Party

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Brendan Lee

Professor and Chairman

Responsibility Role PRINCIPAL_INVESTIGATOR

Principal Investigators

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V. Reid Sutton, M.D.

Role: STUDY_CHAIR

Baylor College of Medicine

Frank Rauch, M.D.

Role: STUDY_CHAIR

McGill University

Locations

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Phoenix Children's Hospital

Phoenix, Arizona, United States

Site Status RECRUITING

University of California Los Angeles

Los Angeles, California, United States

Site Status RECRUITING

AI Dupont Hospital for Children

Wilmington, Delaware, United States

Site Status RECRUITING

Children's National Medical Center

Washington D.C., District of Columbia, United States

Site Status RECRUITING

University of South Florida

Tampa, Florida, United States

Site Status RECRUITING

Kennedy Krieger Institute / Hugo W. Moser Research Institute

Baltimore, Maryland, United States

Site Status RECRUITING

University of Nebraska Medical Center

Omaha, Nebraska, United States

Site Status RECRUITING

Hospital for Special Surgery

New York, New York, United States

Site Status RECRUITING

Oregon Health and Science University

Portland, Oregon, United States

Site Status RECRUITING

Baylor College of Medicine

Houston, Texas, United States

Site Status RECRUITING

Shriners Hospital for Children, Chicago / Marquette University

Milwaukee, Wisconsin, United States

Site Status RECRUITING

Shriners Hospital for Children

Montreal, Quebec, Canada

Site Status RECRUITING

Countries

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United States Canada

Central Contacts

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Dianne Nguyen

Role: CONTACT

713.798.6694

Facility Contacts

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Isabella Wheeler

Role: primary

Sarah Gaunt

Role: primary

Katie Richard

Role: primary

302.298.8367

Andrew Pennington

Role: primary

202-476-5562

Elaine Deval

Role: primary

813-974-0104

Libby Walker

Role: primary

Sarah O'Neill

Role: primary

(402) 616-0562

Chloe DeRocher

Role: primary

Melanie Abrahamson

Role: primary

Dianne Nguyen

Role: primary

713-798-6694

Angela Caudill, MPT

Role: primary

773-385-5868

Michaela Durigova

Role: primary

514-282-7158

Other Identifiers

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H36165

Identifier Type: -

Identifier Source: org_study_id

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