Effects of Bisphosphonates on OI-Related Hearing Loss

April 23, 2026 updated by: Hospital for Special Surgery, New York

Effects of Bisphosphonates on OI-Related Hearing Loss: A Pilot Study

Osteogenesis Imperfecta-related hearing loss usually occurs in individuals with mild (type I) OI and is much earlier in onset than age-related hearing loss, with the majority of individuals experiencing some minor hearing loss in their 20s. Bisphosphonates have been successfully used to treat otosclerosis, a common cause of hearing loss similar to OI-related hearing loss. As many individuals with OI-related hearing loss also present with otosclerosis and because of their mechanistic similarities, the investigators propose studying the effects of bisphosphonate treatment on individuals diagnosed with both OI type I and hearing loss, thereby determining its effectiveness as a potential treatment for hearing loss.

The investigators will enroll 50 individuals diagnosed with type I OI and age 18-100. 25 adults will be enrolled into the treatment arm and receive bisphosphonate treatment (must have at least mild hearing loss), while 25 adults will be enrolled into the control arm. The investigators will enroll 25 children (6-17 years of age) diagnosed with OI who are currently receiving bisphosphonate treatment as part of their care for orthopedic symptoms. The investigators will also observe 25 children (6-17 years of age) diagnosed with OI who are NOT currently receiving bisphosphonate treatment. The study duration is 63 months (approximately 5 years). Enrollment is anticipated to begin in November 2019.

Study Overview

Status

Recruiting

Conditions

Intervention / Treatment

Study Type

Interventional

Enrollment (Estimated)

100

Phase

  • Phase 4

Contacts and Locations

This section provides the contact details for those conducting the study, and information on where this study is being conducted.

Study Contact

Study Contact Backup

  • Name: Erin Carter, MS, CGC
  • Phone Number: (212) 774-7332

Study Locations

  • United States
    • New York
      • New York, New York, United States, 10021
        • Recruiting
        • Hospital for Special Surgery
        • Contact:

Participation Criteria

Researchers look for people who fit a certain description, called eligibility criteria. Some examples of these criteria are a person's general health condition or prior treatments.

Eligibility Criteria

Ages Eligible for Study

6 years to 100 years (Child, Adult, Older Adult)

Accepts Healthy Volunteers

Yes

Description

Inclusion Criteria (Adult Treatment Arm):

  • Diagnosis of OI type I
  • Diagnosis of at least mild hearing loss (>20dB pure tone average) by audiogram testing
  • 18+
  • Vitamin D level > 30

Inclusion Criteria (Adult Control Arm):

  • Diagnosis of OI type I

Inclusion Criteria (Child Observational Bisphosphonate Arm)

  • Diagnosis of OI
  • Age 6-17 years
  • Currently receiving bisphosphonate treatment as standard of care

Inclusion Criteria (Child Observational No Treatment Arm)

  • Diagnosis of OI
  • Age 6-17 years
  • NOT receiving bisphosphonate treatment and will not receive bisphosphonate treatment for the duration of the study

Exclusion Criteria (ALL ARMS):

  • Family history of hearing-loss (not related to OI or occupational hearing loss)
  • Pregnancy

Study Plan

This section provides details of the study plan, including how the study is designed and what the study is measuring.

How is the study designed?

Design Details

  • Primary Purpose: Prevention
  • Allocation: Non-Randomized
  • Interventional Model: Single Group Assignment
  • Masking: None (Open Label)

Arms and Interventions

Participant Group / Arm
Intervention / Treatment
Experimental: Adult Treatment Arm
Intervention treatment arm. Adults (18+ years) with type 1 OI. Must have at least mild hearing loss. Will receive Risedronate (35mg, 0-2x/week as clinically indicated) for duration of study. Changes in hearing, quality of life, and bone density will be monitored.
Drug: Risedronate Oral Tablet
Oral bisphosphonate
Other Names:
  • actonel
No Intervention: Child (Bisphosphonate Arm)
Observational (no investigational intervention) arm. Children (6-17 years) with any type of OI who are already receiving bisphosphonate treatment as standard of care treatment for orthopedic symptoms. Changes in hearing, quality of life, and bone density will be observed for the duration of the study.
No Intervention: Child (Control Arm)
Observational arm. Children (6-17 years) with any type of OI who are not receiving bisphosphonate treatment. Changes in hearing, quality of life, and bone density will be observed for the duration of the study.
No Intervention: Adult Control Arm
Observational arm. Adults (18+ years) with type 1 OI. Changes in hearing, quality of life, and bone density will be monitored.

What is the study measuring?

Primary Outcome Measures

Outcome Measure
Measure Description
Time Frame
Pure Tone Averages
Time Frame: Baseline, 3, 6, 12, 18, 24, 30, 36, 42, 48, 54, 60 months
Average hearing thresholds at 250, 500, 1000, 2000, 3000, 4000, 8000 Hertz
Baseline, 3, 6, 12, 18, 24, 30, 36, 42, 48, 54, 60 months

Secondary Outcome Measures

Outcome Measure
Measure Description
Time Frame
Speech Recognition Scores
Time Frame: Baseline, 3, 6, 12, 18, 24, 30, 36, 42, 48, 54, 60 months
Lowest volume participant can hear and understand speech (decibels)
Baseline, 3, 6, 12, 18, 24, 30, 36, 42, 48, 54, 60 months
Word Recognition Scores
Time Frame: Baseline, 3, 6, 12, 18, 24, 30, 36, 42, 48, 54, 60 months
Percent of words participants correctly repeat in word recognition test (%)
Baseline, 3, 6, 12, 18, 24, 30, 36, 42, 48, 54, 60 months
Hearing Handicap Inventory Raw Score
Time Frame: Yearly (Baseline, 12, 24, 36, 48, 60 months)
Score 0-40. Lower score is better. Adults (self-reported)
Yearly (Baseline, 12, 24, 36, 48, 60 months)
Tinnitus Handicap Inventory Score
Time Frame: Yearly (Baseline, 12, 24, 36, 48, 60 months)
Score 0-100. Lower score is better. Adults (self-reported).
Yearly (Baseline, 12, 24, 36, 48, 60 months)
Dizziness Handicap Inventory Score
Time Frame: Yearly (Baseline, 12, 24, 36, 48, 60 months)
Score 0-100. Lower score is better. Adults (self-reported). Incidence and impact of vertigo in study population.
Yearly (Baseline, 12, 24, 36, 48, 60 months)
SF-36 Scale and Summary Scores
Time Frame: Yearly (Baseline, 12, 24, 36, 48, 60 months)
Score 0-100. Higher score is better. Adults (self-reported) quality-of-life survey.
Yearly (Baseline, 12, 24, 36, 48, 60 months)
Pediatric Outcomes Data Collection Instrument (PODCI) Score
Time Frame: Yearly (Baseline, 12, 24, 36, 48, 60 months)
Score 0-100. Lower score is better. Children (ages 6-10 years), parent-reported. Assessment of overall health and functioning.
Yearly (Baseline, 12, 24, 36, 48, 60 months)
Adolescent Outcomes Questionnaire Score
Time Frame: Yearly (Baseline, 12, 24, 36, 48, 60 months)
Score 0-100. Lower score is better. Children (ages 11-17 years), parent- or self-reported. Assessment of overall health and functioning.
Yearly (Baseline, 12, 24, 36, 48, 60 months)
DEXA Z-score
Time Frame: Yearly (Baseline, 12, 24, 36, 48, 60 months)
Higher score is better. Relative Bone Density
Yearly (Baseline, 12, 24, 36, 48, 60 months)
DEXA Bone Mineral Density
Time Frame: Yearly (Baseline, 12, 24, 36, 48, 60 months)
Higher score is better. Bone Mineral Density (grams/centimeter^2)
Yearly (Baseline, 12, 24, 36, 48, 60 months)

Collaborators and Investigators

This is where you will find people and organizations involved with this study.

Sponsor

Hospital for Special Surgery, New York

Collaborators

Northwell Health
The New York Community Trust
East River Medical Imaging

Investigators

  • Principal Investigator: Cathleen Raggio, MD, Hospital for Special Surgery, New York

Publications and helpful links

The person responsible for entering information about the study voluntarily provides these publications. These may be about anything related to the study.

General Publications

Study record dates

These dates track the progress of study record and summary results submissions to ClinicalTrials.gov. Study records and reported results are reviewed by the National Library of Medicine (NLM) to make sure they meet specific quality control standards before being posted on the public website.

Study Major Dates

Study Start (Actual)

November 2, 2019

Primary Completion (Estimated)

January 1, 2027

Study Completion (Estimated)

January 1, 2027

Study Registration Dates

First Submitted

October 18, 2019

First Submitted That Met QC Criteria

November 1, 2019

First Posted (Actual)

November 5, 2019

Study Record Updates

Last Update Posted (Actual)

April 28, 2026

Last Update Submitted That Met QC Criteria

April 23, 2026

Last Verified

January 1, 2026

More Information

Terms related to this study

Keywords

Additional Relevant MeSH Terms

Other Study ID Numbers

  • #2018-0700

Plan for Individual participant data (IPD)

Plan to Share Individual Participant Data (IPD)?

NO

Drug and device information, study documents

Studies a U.S. FDA-regulated drug product

Yes

Studies a U.S. FDA-regulated device product

No

product manufactured in and exported from the U.S.

Yes

This information was retrieved directly from the website clinicaltrials.gov without any changes. If you have any requests to change, remove or update your study details, please contact register@clinicaltrials.gov. As soon as a change is implemented on clinicaltrials.gov, this will be updated automatically on our website as well.

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