MedPath

Valganciclovir Therapy in Infants and Children With Congenital CMV Infection and Hearing Loss

Phase 2
Completed
Conditions
Cytomegalovirus Infection
Interventions
Other: Placebo
Registration Number
NCT01649869
Lead Sponsor
National Institute of Allergy and Infectious Diseases (NIAID)
Brief Summary

This is an international, multi-center, double-blind, placebo-controlled evaluation valganciclovir treatment for up to 54 children (up to 4 years of age) with virologically-confirmed congenital CMV infection and hearing loss. Subject participation will be over a six-month period and study subjects will be stratified according to age. The primary objective is to assess whether a six-week course of oral valganciclovir can stabilize the hearing of children with congenital CMV infection who present with hearing loss.

Detailed Description

Congenital cytomegalovirus (CMV) infection is the most frequent known viral cause of mental retardation, and is the leading non-genetic cause of sensorineural hearing loss in many countries including the United States. This is a Phase II international, multi-center, double-blind, placebo-controlled evaluation of 6 weeks of oral valganciclovir treatment or 6 weeks of placebo for fifty-four male and female infants/toddlers 1 month through 3 years of age (up to 4 years of age) with virologically-confirmed congenital CMV infection and hearing loss. Patient who are between 1 month and 4 years of age and who have SNHL (Sensorineural Hearing Loss) and are eligible for enrollment. The expected study duration is 3.5 years from enrollment of first study subject. The primary objective is to assess whether a six week course of oral valganciclovir can stabilize the hearing of children with congenital CMV infection who present with hearing loss. The secondary objective is to define the following responses as a function of systemic exposure to ganciclovir (active metabolite of valganciclovir): CMV viral load in blood; CMV viral load in urine; and CMV viral load in saliva. Also, to define the safety and tolerability of valganciclovir in enrolled subjects. The tertiary objective is to define the pharmacokinetics of ganciclovir (metabolite) following administration of valganciclovir (prodrug) in enrolled subjects.

Recruitment & Eligibility

Status
COMPLETED
Sex
All
Target Recruitment
54
Inclusion Criteria
  1. Signed informed consent from parent(s) or legal guardian(s)
  2. Sensorineural hearing loss (>/= 21dB in one or both ears, documented within 12 weeks prior to study entry)
  3. Children from 1 month through 3 years of age (up to the 4th birthday)
Exclusion Criteria
  1. Imminent demise
  2. Profound sensorineural hearing loss (> 90dB) in both ears
  3. Patients receiving other antiviral agents or immune globulin
  4. Gastrointestinal abnormality which might preclude absorption of an oral medication (e.g., a history of necrotizing enterocolitis)
  5. Documented renal insufficiency, as noted by a creatinine clearance < 10 mL/min/1.73m2 at time of study enrollment
  6. Breastfeeding from mother who is receiving ganciclovir, valganciclovir, foscarnet, cidofovir, or maribavir
  7. Infants known to be born to women who are HIV positive (but HIV testing is not required for study entry).
  8. Current receipt of other investigational drugs
  9. Previous receipt of ganciclovir or valganciclovir
  10. Known hypersensitivity to ganciclovir, valganciclovir, or components of the product
  11. Inability to attend follow-up hearing and clinical assessments
  12. Infants with Auditory neuropathy/dyssynchrony.
  13. Children with another known etiology for SNHL (e.g. connexin 26, syndrome or metabolic disorder associated with SNHL, inner ear malformation and widened vestibular aqueducts, meningitis).

Exclusion of each of these conditions is not required for trial enrollment.

Study & Design

Study Type
INTERVENTIONAL
Study Design
PARALLEL
Arm && Interventions
GroupInterventionDescription
PlaceboPlacebo27 Children between 1 month and 3 years of age (up to 4th birthday) with sensoneural hearing loss and documented CMV infection will receive placebo orally twice a day for 6 weeks
ActiveValganciclovir27 Children between 1 month and 3 years of age (up to 4th birthday) with sensoneural hearing loss and documented CMV infection will receive valganciclovir HCl 16.0 mg/kg orally twice a day for 6 weeks
Primary Outcome Measures
NameTimeMethod
Number of Ears That Had (1) Improved Hearing or no Change in Hearing (2) Worsened Hearing.Day 1 through Day 180

A single, independent study audiologist who is masked (blinded) to treatment assignment will assess the audiology test battery for each subject and assign the classifications of normal hearing, mild hearing loss, moderate hearing loss, or severe hearing loss based upon their hearing thresholds (in decibels). The classifications will be assigned by ear (one for the left ear and one for the right ear), giving "total ear" classifications. At the analyses stage, the "best ear" classification for the subject at that study visit will be determined; for example, if a subject had mild hearing loss in their left ear and severe hearing loss in their right ear, then the "best ear" classification will be mild hearing loss. Not both ears are evaluable for all subjects. In some subjects, only one ear is evaluable.

Secondary Outcome Measures
NameTimeMethod
Association of Change in Viral Load (Saliva) With Change in Total Ear Hearing at 6 MonthsAt 6 months

Analysis of actual viral load was done using log base 10 transformation. Undetectable viral load value was replaced by a value of 10. A summary measure of the viral load over time considers all time points available by calculating the average area under the curve (AUC) (trapezoidal rule) applied to the log base 10 viral load. Average is based on the maximum period of time with viral load data for a given subject. The average or standardize AUC units is therefore the original AUC units or log 10 copies/ml\*days divided by days in study which equals log 10 copies/ml.

Change in Best Ear Hearing Assessments [Worse + no Change (Abnormal to Abnormal) Versus Other] Between Baseline and Study Month 6.Day 1 through Day 180

A single, independent study audiologist who is masked (blinded) to treatment assignment will assess the audiology test battery for each subject and assign the classifications of normal hearing, mild hearing loss, moderate hearing loss, or severe hearing loss based upon their hearing thresholds (in decibels). The classifications will be assigned by ear (one for the left ear and one for the right ear), giving "total ear" classifications. At the analyses stage, the "best ear" classification for the subject at that study visit will be determined; for example, if a subject had mild hearing loss in their left ear and severe hearing loss in their right ear, then the "best ear" classification will be mild hearing loss.

Number of Best Ear That Had (1) Improved Hearing or no Change in Hearing (2) Worsened Hearing [ex. Improved+ no Change (Normal to Normal) Versus Other].Day 1 through Day 180

A single, independent study audiologist who is masked (blinded) to treatment assignment will assess the audiology test battery for each subject and assign the classifications of normal hearing, mild hearing loss, moderate hearing loss, or severe hearing loss based upon their hearing thresholds (in decibels). The classifications will be assigned by ear (one for the left ear and one for the right ear), giving "total ear" classifications. At the analyses stage, the "best ear" classification for the subject at that study visit will be determined; for example, if a subject had mild hearing loss in their left ear and severe hearing loss in their right ear, then the "best ear" classification will be mild hearing loss. For this outcome, we combine the improved hearing and no change for the special case only of normal to normal. Other category include worsened and no change from (1) mild to mild hearing loss, (2) moderate to moderate hearing loss, or (3) severe to severe hearing loss.

Change in Best Ear Hearing Assessments [Improved Versus Other] Between Baseline and Study Month 6.Day 1 through Day 180

A single, independent study audiologist who is masked (blinded) to treatment assignment will assess the audiology test battery for each subject and assign the classifications of normal hearing, mild hearing loss, moderate hearing loss, or severe hearing loss based upon their hearing thresholds (in decibels). The classifications will be assigned by ear (one for the left ear and one for the right ear), giving "total ear" classifications. At the analyses stage, the "best ear" classification for the subject at that study visit will be determined; for example, if a subject had mild hearing loss in their left ear and severe hearing loss in their right ear, then the "best ear" classification will be mild hearing loss.

Change in Best Ear Hearing Assessments [Worse Versus Other] Between Baseline and Study Month 6.Day 1 through Day 180

A single, independent study audiologist who is masked (blinded) to treatment assignment will assess the audiology test battery for each subject and assign the classifications of normal hearing, mild hearing loss, moderate hearing loss, or severe hearing loss based upon their hearing thresholds (in decibels). The classifications will be assigned by ear (one for the left ear and one for the right ear), giving "total ear" classifications. At the analyses stage, the "best ear" classification for the subject at that study visit will be determined; for example, if a subject had mild hearing loss in their left ear and severe hearing loss in their right ear, then the "best ear" classification will be mild hearing loss.

Association of Change in Viral Load (Blood) With Change in Best Ear Hearing at 6 MonthsAt 6 months

Analysis of actual viral load was done using log base 10 transformation. Undetectable viral load value was replaced by a value of 10. A summary measure of the viral load over time considers all time points available by calculating the average area under the curve (AUC) (trapezoidal rule) applied to the log base 10 viral load. Average is based on the maximum period of time with viral load data for a given subject. The average or standardize AUC units is therefore the original AUC units of log 10 copies/ml\*days divided by days in study which equals log 10 copies/ml.

Association of Change in Viral Load (Saliva) With Change in Best Ear Hearing at 6 MonthsAt 6 months

Analysis of actual viral load was done using log base 10 transformation. Undetectable viral load value was replaced by a value of 10. A summary measure of the viral load over time considers all time points available by calculating the average area under the curve (AUC) (trapezoidal rule) applied to the log base 10 viral load. Average is based on the maximum period of time with viral load data for a given subject. The average or standardize AUC units is therefore the original AUC units of log 10 copies/ml\*days divided by days in study which equals log 10 copies/ml.

Change in Total Ear Hearing Assessments [Improved Versus Other] Between Baseline and Study Month 6.Day 1 through Day 180

A single, independent study audiologist who is masked (blinded) to treatment assignment will assess the audiology test battery for each subject and assign the classifications of normal hearing, mild hearing loss, moderate hearing loss, or severe hearing loss based upon their hearing thresholds (in decibels). The classifications will be assigned by ear (one for the left ear and one for the right ear), giving "total ear" classifications. At the analyses stage, the "best ear" classification for the subject at that study visit will be determined; for example, if a subject had mild hearing loss in their left ear and severe hearing loss in their right ear, then the "best ear" classification will be mild hearing loss.

Association of Change in Viral Load (Blood) With Change in Total Ear Hearing at 6 MonthsAt 6 months

Analysis of actual viral load was done using log base 10 transformation. Undetectable viral load value was replaced by a value of 10. A summary measure of the viral load over time considers all time points available by calculating the average area under the curve (AUC) (trapezoidal rule) applied to the log base 10 viral load. Average is based on the maximum period of time with viral load data for a given subject. The average or standardize AUC units is therefore the original AUC units of log 10 copies/ml\*days divided by days in study which equals log 10 copies/ml.

Detection of Viremia (Blood) by PCR Six Weeks After Trial EntryAt 6 weeks (Day 42)

Each subject either has positive or negative PCR results. Virus is detected if the PCR is positive.

Detection of CMV in Saliva PCR Six Month After Trial EntryAt 6 months

Each subject either has positive or negative PCR results. Virus is detected if the PCR is positive.

The Quantitative Log Reduction in Viruria Detected After 6 Weeks of TherapyBaseline thru months 6

The quantitative log reduction in viruria (urine) detected after 6 weeks of therapy. Quantitative viral load by PCR in log 10 units measured in urine after 6 weeks of therapy; if undetectable, viral load is assigned a value of 10 (1 in log 10 units)

Change in Total Ear Hearing Assessments [Worse Versus Other] Between Baseline and Study Month 6.Day 1 through Day 180

A single, independent study audiologist who is masked (blinded) to treatment assignment will assess the audiology test battery for each subject and assign the classifications of normal hearing, mild hearing loss, moderate hearing loss, or severe hearing loss based upon their hearing thresholds (in decibels). The classifications will be assigned by ear (one for the left ear and one for the right ear), giving "total ear" classifications. At the analyses stage, the "best ear" classification for the subject at that study visit will be determined; for example, if a subject had mild hearing loss in their left ear and severe hearing loss in their right ear, then the "best ear" classification will be mild hearing loss.

Change in Total Ear Hearing Assessments [Worse+ no Change (Abnormal to Abnormal) Versus Other] Between Baseline and Study Month 6.Day 1 through Day 180

A single, independent study audiologist who is masked (blinded) to treatment assignment will assess the audiology test battery for each subject and assign the classifications of normal hearing, mild hearing loss, moderate hearing loss, or severe hearing loss based upon their hearing thresholds (in decibels). The classifications will be assigned by ear (one for the left ear and one for the right ear), giving "total ear" classifications. At the analyses stage, the "best ear" classification for the subject at that study visit will be determined; for example, if a subject had mild hearing loss in their left ear and severe hearing loss in their right ear, then the "best ear" classification will be mild hearing loss.

Detection of CMV in Saliva by PCR Six Weeks After Trial EntryAt 6 weeks (Day 42)

Each subject either has positive or negative PCR results. Virus is detected if the PCR is positive.

The Quantitative Log Change in Viremia From Baseline to Month 6.Baseline to month 6

The quantitative change (Month 6 minus baseline) in viremia (blood) Quantitative viral load by PCR in log 10 units measured in urine after 6 weeks of therapy; if undetectable, viral load is assigned a value of 10 (1 in log 10 units).

Association of Change in Viral Load (Urine) With Change in Total Ear Hearing at 6 MonthsAt 6 months

Analysis of actual viral load was done using log base 10 transformation. Undetectable viral load value was replaced by a value of 10. A summary measure of the viral load over time considers all time points available by calculating the average area under the curve (AUC) (trapezoidal rule) applied to the log base 10 viral load. Average is based on the maximum period of time with viral load data for a given subject. The average or standardize AUC units is therefore the original AUC units of log 10 copies/ml\*days divided by days in study which equals log 10 copies/ml.

Detection of Viruria (Urine) by PCR Six Weeks After Trial EntryAt 6 weeks (Day 42)

Each subject either has positive or negative PCR results. Virus is detected if the PCR is positive.

Detection of Viruria (Urine) by PCR Six Month After Trial EntryAt 6 months

Each subject either has positive or negative PCR results. Virus is detected if the PCR is positive.

Detection of Viremia (Blood) by PCR Six Month After Trial EntryAt 6 months

Each subject either has positive or negative PCR results. Virus is detected if the PCR is positive.

Adverse Event (AE) Resulting in Unanticipated Medically Attended VisitDay 1 thru day 70

Adverse event resulting in unanticipated medically attended visit. This outcome summarizes the number of adverse events (AEs) that resulted in the unanticipated medically attended visit.

Association of Change in Viral Load (Urine) With Change in Best Ear Hearing at 6 MonthsAt 6 months

Analysis of actual viral load was done using log base 10 transformation. Undetectable viral load value was replaced by a value of 10. A summary measure of the viral load over time considers all time points available by calculating the average area under the curve (AUC) (trapezoidal rule) applied to the log base 10 viral load. Average is based on the maximum period of time with viral load data for a given subject. The average or standardize AUC units is therefore the original AUC units of log 10 copies/ml\*days divided by days in study which equals log 10 copies/ml.

Adverse Event (AE) Resulting in Unresolved OutcomeDay 1 thru day 70

Adverse event resulting in unresolved outcome. This outcome summarizes the number of adverse events (AEs) that resulted in unresolved outcome of that AE.

The Quantitative Log Reduction in CMV in Saliva Detected After 6 Weeks of TherapyBaseline thru months 6

The quantitative log reduction in CMV in saliva (urine) detected after 6 weeks of therapy. Quantitative viral load by PCR in log 10 units measured in urine after 6 weeks of therapy; if undetectable, viral load is assigned a value of 10 (1 in log 10 units)

Number of Adverse Events in the Active Group That Resulted in Discontinuation of ValganciclovirDay 1 thru day 70

AE resulting in discontinuation of valganciclovir (active group only). This outcome summarizes the number of adverse events (AEs) that resulted in the discontinuation of valganciclovir in the active group only.

Trial Locations

Locations (18)

Children's of Alabama Child Health Research Unit (CHRU)

🇺🇸

Birmingham, Alabama, United States

University of Rochester Medical Center

🇺🇸

Rochester, New York, United States

Children's National Medical Center - Sheikh Zayed Campus - Infectious Disease

🇺🇸

Washington, District of Columbia, United States

Washington University School of Medicine in St. Louis - Center for Clinical Studies

🇺🇸

Saint Louis, Missouri, United States

Steven and Alexandra Cohen Childrens Medical Center of New York - New Hyde Park - Infectious Disease

🇺🇸

Manhasset, New York, United States

Carolinas Medical Center - Pediatrics - Infectious Diseases

🇺🇸

Charlotte, North Carolina, United States

Nationwide Children's Hospital - Neonatology - Center for Perinatal Research

🇺🇸

Columbus, Ohio, United States

Rhode Island Hospital - Pediatrics

🇺🇸

Providence, Rhode Island, United States

Texas Medical Center - Texas Children's Hospital

🇺🇸

Houston, Texas, United States

Bristol Royal Hospital for Children - Paediatric Immunology

🇬🇧

Bristol, Bristol, City Of, United Kingdom

Saint George's Hospital - Pediatric Infectious Diseases

🇬🇧

London, London, City Of, United Kingdom

Great Ormond Street Hospital - Infectious Diseases

🇬🇧

London, London, City Of, United Kingdom

Birmingham Heartlands Hospital

🇬🇧

Birmingham, United Kingdom

Pennine Acute Hospitals NHS Trust, North Manchester General Hospital - Children's and Adolescent Services

🇬🇧

Crumpsall, United Kingdom

John Radcliffe Hospital - Children's Hospital - Paediatric Infectious Disease and Immunology

🇬🇧

Headington, Oxford, United Kingdom

The Great North Children's Hospital - Paediatric Immunology

🇬🇧

Newcastle Upon Tyne, United Kingdom

Sheffield Children's Hospital - Immunology

🇬🇧

Sheffield, United Kingdom

Southampton Children's Hospital - Allergy, Immunology and Infection

🇬🇧

Southampton, Hampshire, United Kingdom

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