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Neurobiology and Treatment of Reading Disability in NF-1

This study has been completed.
Sponsor:
Collaborator:
National Institute of Neurological Disorders and Stroke (NINDS)
Information provided by (Responsible Party):
Laura Cutting, Vanderbilt University
ClinicalTrials.gov Identifier:
NCT00624234
First received: February 25, 2008
Last updated: April 11, 2017
Last verified: April 2017
  Purpose
The goal of this trial is to determine if children with neurofibromatosis type 1 who have reading disabilities respond the same way—both behaviorally and neurobiologically—to specialized treatment programs as children with idiopathic reading disabilities do, and to determine which intervention is best for particular learner profiles.

Condition Intervention
Neurofibromatosis Type 1
Reading Disabilities
Behavioral: Tutoring Program I
Behavioral: Tutoring Program II

Study Type: Interventional
Study Design: Allocation: Randomized
Intervention Model: Parallel Assignment
Masking: Participant
Primary Purpose: Treatment
Official Title: Neurobiology and Treatment of Reading Disability in Neurofibromatosis Type 1 (NF-1)

Resource links provided by NLM:


Further study details as provided by Vanderbilt University:

Primary Outcome Measures:
  • Change From Baseline in WJ-III Basic Reading Normative Update (Woodcock Johnson Psychoeducational Battery - 3rd Edition; WJ-III NU) Standard Score at 15 Hours [ Time Frame: 0 and 15 hours ]
    This metric measures change in reading abilities, including word recognition and decoding, as assessed by standard educational assessments (Woodcock Johnson Psychoeducational Battery - 3rd Edition Normative Update; WJ-III NU). The scores are reported as change in age-normed standard scores (a change of 15 standard score points would represent a change of 1 standard deviation in the general population).The Basic Reading score is a normed composite of the WJ-III subtests Letter-Word Identification and Word Attack, representing word-level reading skill.


Secondary Outcome Measures:
  • Neuroimaging Data [ Time Frame: Collected before and after intervention ]
    Neuroimaging data consists of functional MRI and structural MRI measures.


Enrollment: 184
Actual Study Start Date: February 2006
Study Completion Date: January 2017
Primary Completion Date: March 2013 (Final data collection date for primary outcome measure)
Arms Assigned Interventions
Experimental: NF-Tutoring Program 1
Tutoring Program I
Behavioral: Tutoring Program I
Tutoring Program I is a structured multi-sensory program that is designed to gradually present the range of sounds and letters with focus on accuracy of phonological concepts and application of those concepts in phrases and sentences. The instruction uses a sequenced defined lesson plan with accuracy and automaticity criteria for skill progression. A range of manipulative and kinesthetic activities is outlined to maintain learner engagement in the intensive intervention design.
Experimental: NF-Tutoring Program 2
Tutoring Program II
Behavioral: Tutoring Program II
Tutoring Program II is designed to teach visual and speech elements of reading separately at first, and then bring them together for maximum efficiency. The program uses the idea of teaching concepts about the structure of words. For example, students transfer the rules they have learned about one vowel or structure to another without specific instructions on the new one. Tutoring Program II incorporates pictures and activities to help remember strategies for increasing basic reading skills. Speed drills are also used for development of decoding automaticity.
No Intervention: Typically Developing Readers
Control group
Experimental: IRD-Tutoring Program 1
Tutoring Program I
Behavioral: Tutoring Program I
Tutoring Program I is a structured multi-sensory program that is designed to gradually present the range of sounds and letters with focus on accuracy of phonological concepts and application of those concepts in phrases and sentences. The instruction uses a sequenced defined lesson plan with accuracy and automaticity criteria for skill progression. A range of manipulative and kinesthetic activities is outlined to maintain learner engagement in the intensive intervention design.
Experimental: IRD-Tutoring Program 2
Tutoring Program II
Behavioral: Tutoring Program II
Tutoring Program II is designed to teach visual and speech elements of reading separately at first, and then bring them together for maximum efficiency. The program uses the idea of teaching concepts about the structure of words. For example, students transfer the rules they have learned about one vowel or structure to another without specific instructions on the new one. Tutoring Program II incorporates pictures and activities to help remember strategies for increasing basic reading skills. Speed drills are also used for development of decoding automaticity.
No Intervention: Waitlist Control
Intervention Control Group (RD)

Detailed Description:

The most common concern of parents of children with neurofibromatosis type 1 (NF-1) is learning disabilities (LD). Approximately one half of all children with NF-1 have LD—the most debilitating and common of which are reading disabilities.

The purpose of this study is to determine if children with NF-1 who have reading disabilities respond the same way—both behaviorally and neurobiologically—to specialized treatment programs known to improve the decoding deficits in children with idiopathic reading disabilities. The trial will also determine which intervention is best for particular learner profiles. The overall purpose of this research is to gain a deeper understanding of the characteristics and treatment of reading disabilities in NF-1.

In the trial, researchers will compare children with NF-1 who show weaknesses in reading to children with reading disabilities of no known cause (idiopathic) using two different interventions and behavioral and neurobiological measures. Both interventions focus on teaching sound-symbol relationships, but vary in terms of relative emphasis on verbal versus visual methods of teaching.

Scientists hope findings from the trial will advance knowledge about the best therapies for LD in children with NF-1. And, by further refining how children with NF-1 who have reading disabilities are similar (or different) to children with idiopathic reading disabilities, the researchers may be able to learn if reading interventions that address areas other than decoding will also benefit children with NF-1. Also, by understanding the similarities and/or differences in the neuropsychological and neurobiological profiles of children with NF-1 who have reading disabilities, and those without, scientists will be able to refine the cognitive phenotype and neurobiological characteristics of NF-1, which will further understanding of central nervous system abnormalities in NF-1.

  Eligibility

Ages Eligible for Study:   8 Years to 17 Years   (Child)
Sexes Eligible for Study:   All
Accepts Healthy Volunteers:   Yes
Criteria

Inclusion Criteria:

This study will be open to all individuals, ages 8 to 17 years, who meet eligibility criteria regardless of race, gender, or socioeconomic status.

  • The Reading Disabilities group (including those with NF-1) is defined by scoring equal to or less than the 25th percentile on measures of basic word reading skills.
  • The Control group (including those with NF1) is defined by scoring equal to or above the 40th percentile on the average of the Letter Word Identification and Word Attack subtests from the WJ-III.

Exclusion Criteria:

Any child, regardless of which group he/she is recruited for, will be excluded if he/she meets any of the following criteria (determined during phone screening, medical review, and during testing):

  • is in foster care;
  • previous diagnosis of mental retardation;
  • known uncorrectable visual impairment;
  • history of known neurological disorder (e.g., epilepsy, spina bifida, cerebral palsy, traumatic brain injury);
  • documented hearing impairment greater than 25 dB loss in either ear;
  • medical contraindication to MRI procedures, if participating in MRI (including exposure to metal and pregnancy);
  • individuals known to have an IQ below 70;
  • history or presence of a pervasive developmental disorder;
  • during the DICA-IV parents indicate the presence of any severe psychiatric diagnoses or pervasive developmental disorder.
  Contacts and Locations
Choosing to participate in a study is an important personal decision. Talk with your doctor and family members or friends about deciding to join a study. To learn more about this study, you or your doctor may contact the study research staff using the Contacts provided below. For general information, see Learn About Clinical Studies.

Please refer to this study by its ClinicalTrials.gov identifier: NCT00624234

Locations
United States, Tennessee
Vanderbilt University, Vanderbilt University Institute of Imaging Science
Nashville, Tennessee, United States, 37232-2310
Sponsors and Collaborators
Vanderbilt University
National Institute of Neurological Disorders and Stroke (NINDS)
Investigators
Principal Investigator: Laurie E. Cutting, Ph.D. Vanderbilt University Special Education Department
Principal Investigator: Martha Bridge Denckla, M.D. Kennedy Krieger Institute, Johns Hopkins University, Johns Hopkins University School of Medicine
Principal Investigator: Sheryl L. Rimrodt, M.D. Vanderbilt University Pediatrics Department
Principal Investigator: John Gore, Ph.D. Vanderbilt University
  More Information

Additional Information:
Responsible Party: Laura Cutting, Associate Professor, Vanderbilt University
ClinicalTrials.gov Identifier: NCT00624234     History of Changes
Other Study ID Numbers: R01NS049096 ( US NIH Grant/Contract Award Number )
NF1-R01
Study First Received: February 25, 2008
Results First Received: August 5, 2016
Last Updated: April 11, 2017
Individual Participant Data  
Plan to Share IPD: Yes
Plan Description: De-identified behavioral data (Woodcock Johnson-III NU scores) may be shared with other researchers upon request. De-identified T1 structural neuroimaging scans will be shared with other researchers through a neuroimaging data warehouse or consortium.

Keywords provided by Vanderbilt University:
neurofibromatosis type 1
reading disabilities
NF-1
learning disabilities
tutoring intervention

Additional relevant MeSH terms:
Neurofibromatoses
Neurofibroma
Neurofibromatosis 1
Dyslexia
Nerve Sheath Neoplasms
Neoplasms, Nerve Tissue
Neoplasms by Histologic Type
Neoplasms
Neoplastic Syndromes, Hereditary
Neurocutaneous Syndromes
Nervous System Diseases
Heredodegenerative Disorders, Nervous System
Neurodegenerative Diseases
Genetic Diseases, Inborn
Peripheral Nervous System Neoplasms
Nervous System Neoplasms
Peripheral Nervous System Diseases
Neuromuscular Diseases
Language Disorders
Communication Disorders
Neurobehavioral Manifestations
Neurologic Manifestations
Learning Disorders
Signs and Symptoms
Neurodevelopmental Disorders
Mental Disorders

ClinicalTrials.gov processed this record on April 24, 2017