Study of the Pathogenesis of Rett Syndrome
Primary Purpose
Rett Syndrome
Status
Completed
Phase
Not Applicable
Locations
Study Type
Interventional
Intervention
dextromethorphan
topiramate
Donepezil
Sponsored by
About this trial
This is an interventional treatment trial for Rett Syndrome focused on measuring Rett syndrome, neurologic and psychiatric disorders, rare disease
Eligibility Criteria
PROTOCOL ENTRY CRITERIA: Rett syndrome Diagnosis confirmed on study
Sites / Locations
Outcomes
Primary Outcome Measures
Secondary Outcome Measures
Full Information
NCT ID
NCT00004807
First Posted
February 24, 2000
Last Updated
June 23, 2005
Sponsor
Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD)
Collaborators
Johns Hopkins University
1. Study Identification
Unique Protocol Identification Number
NCT00004807
Brief Title
Study of the Pathogenesis of Rett Syndrome
Study Type
Interventional
2. Study Status
Record Verification Date
December 1998
Overall Recruitment Status
Completed
Study Start Date
January 1995 (undefined)
Primary Completion Date
undefined (undefined)
Study Completion Date
undefined (undefined)
3. Sponsor/Collaborators
Name of the Sponsor
Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD)
Collaborators
Johns Hopkins University
4. Oversight
5. Study Description
Brief Summary
OBJECTIVES: I. Extend current knowledge of the phenotype and natural history of Rett syndrome (RS).
II. Continue the search for a cytogenetic and/or DNA marker. III. Study the effects of cholinergic drugs based on preliminary evidence for reduced levels of brain acetylcholine, while continuing supportive care to modify seizures, respiratory abnormalities, and motor disturbances, and improve nutrition, behavior, and learning.
IV. Identify targets for future therapeutic interventions, e.g., growth factors, to influence neurologic recovery.
Detailed Description
PROTOCOL OUTLINE: Patients receive a comprehensive clinical evaluation including an ophthalmologic exam; speech, communication, and developmental assessment; nutritional evaluation; neurologic exam; respiratory monitoring; and cytogenetic studies.
Selected patients with malnutrition are given night feedings of Pediasure with Fiber by nasogastric tube. Specific recommendations for feeding techniques and diet are made.
Selected patients with seizures or severe hyperventilation and progressive rigidity are nonrandomly assigned to dextromethorphan or topiramate therapy.
Oral dextromethorphan is maintained 6 months to 1 year; duration of therapy depends on response. Oral topiramate is given for 6 months to 1 year, and Aricept for 6 months to 1 year.
Concurrent anticonvulsants may require dose adjustments while on above protocols. Supportive care for constipation, scoliosis, and weight loss is allowed.
6. Conditions and Keywords
Primary Disease or Condition Being Studied in the Trial, or the Focus of the Study
Rett Syndrome
Keywords
Rett syndrome, neurologic and psychiatric disorders, rare disease
7. Study Design
Primary Purpose
Treatment
Study Phase
Not Applicable
Enrollment
120 (false)
8. Arms, Groups, and Interventions
Intervention Type
Drug
Intervention Name(s)
dextromethorphan
Intervention Type
Drug
Intervention Name(s)
topiramate
Intervention Type
Drug
Intervention Name(s)
Donepezil
10. Eligibility
Sex
All
Minimum Age & Unit of Time
0 Years
Maximum Age & Unit of Time
45 Years
Accepts Healthy Volunteers
No
Eligibility Criteria
PROTOCOL ENTRY CRITERIA:
Rett syndrome
Diagnosis confirmed on study
Overall Study Officials:
First Name & Middle Initial & Last Name & Degree
Sakkubai Naidu
Organizational Affiliation
Johns Hopkins University
Official's Role
Study Chair
12. IPD Sharing Statement
Citations:
PubMed Identifier
1658685
Citation
Wenk GL, Naidu S, Casanova MF, Kitt CA, Moser H. Altered neurochemical markers in Rett's syndrome. Neurology. 1991 Nov;41(11):1753-6. doi: 10.1212/wnl.41.11.1753.
Results Reference
background
PubMed Identifier
8338347
Citation
Reiss AL, Faruque F, Naidu S, Abrams M, Beaty T, Bryan RN, Moser H. Neuroanatomy of Rett syndrome: a volumetric imaging study. Ann Neurol. 1993 Aug;34(2):227-34. doi: 10.1002/ana.410340220.
Results Reference
background
PubMed Identifier
8403377
Citation
Wenk GL, O'Leary M, Nemeroff CB, Bissette G, Moser H, Naidu S. Neurochemical alterations in Rett syndrome. Brain Res Dev Brain Res. 1993 Jul 16;74(1):67-72. doi: 10.1016/0165-3806(93)90084-n.
Results Reference
background
PubMed Identifier
7607612
Citation
Cooke DW, Naidu S, Plotnick L, Berkovitz GD. Abnormalities of thyroid function and glucose control in subjects with Rett syndrome. Horm Res. 1995;43(6):273-8. doi: 10.1159/000184309.
Results Reference
background
PubMed Identifier
7566454
Citation
Naidu S, Hyman S, Harris EL, Narayanan V, Johns D, Castora F. Rett syndrome studies of natural history and search for a genetic marker. Neuropediatrics. 1995 Apr;26(2):63-6. doi: 10.1055/s-2007-979724.
Results Reference
background
PubMed Identifier
2344026
Citation
Naidu S, Hyman S, Piazza K, Savedra J, Perman J, Wenk G, Kitt C, Troncoso J, Price D, Cassanova M, et al. The Rett syndrome: progress report on studies at the Kennedy Institute. Brain Dev. 1990;12(1):5-7. doi: 10.1016/s0387-7604(12)80164-1.
Results Reference
background
PubMed Identifier
7580176
Citation
Casanova MF, Naidu S, Goldberg TE, Moser HW, Khoromi S, Kumar A, Kleinman JE, Weinberger DR. Quantitative magnetic resonance imaging in Rett syndrome. J Neuropsychiatry Clin Neurosci. 1991 Winter;3(1):66-72. doi: 10.1176/jnp.3.1.66.
Results Reference
background
PubMed Identifier
1385677
Citation
Naidu S, Wong DF, Kitt C, Wenk G, Moser HW. Positron emission tomography in the Rett syndrome: clinical, biochemical and pathological correlates. Brain Dev. 1992 May;14 Suppl:S75-9.
Results Reference
background
PubMed Identifier
8040765
Citation
Marcus CL, Carroll JL, McColley SA, Loughlin GM, Curtis S, Pyzik P, Naidu S. Polysomnographic characteristics of patients with Rett syndrome. J Pediatr. 1994 Aug;125(2):218-24. doi: 10.1016/s0022-3476(94)70196-2.
Results Reference
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Study of the Pathogenesis of Rett Syndrome
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