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Neurobiology and Treatment of Reading Disability in NF-1

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ClinicalTrials.gov Identifier: NCT00624234
Recruitment Status : Completed
First Posted : February 27, 2008
Results First Posted : April 10, 2017
Last Update Posted : November 8, 2018
Sponsor:
Collaborator:
National Institute of Neurological Disorders and Stroke (NINDS)
Information provided by (Responsible Party):
Laura Cutting, Vanderbilt University

Study Type Interventional
Study Design Allocation: Randomized;   Intervention Model: Parallel Assignment;   Masking: Single (Participant);   Primary Purpose: Treatment
Conditions Neurofibromatosis Type 1
Reading Disabilities
Interventions Behavioral: Tutoring Program I
Behavioral: Tutoring Program II
Enrollment 184
Recruitment Details Recruitment period was 2006-2012 and was done via NF organizations and NF clinics, as well as the community recruitment.
Pre-assignment Details Exclusions: Incomplete Data (n=31), NF without Reading Difficulty (n=11), Age (n=13; excluded CNT and RD age 15yr and over because no NF-RD over 14yr), Low IQ (n=2), Ineligible (n=12; did not fit criteria for either RD or CNT group), Prior to Intervention Change (n=26; enrolled prior to an NIH-approved change in study design regarding intervention)
Arm/Group Title NF-Tutoring Program 1 NF-Tutoring Program 2 Typically Developing Readers IRD- Tutoring Program 1 IRD-Tutoring Program 2 Waitlist Control
Hide Arm/Group Description

Tutoring Program I

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.

Tutoring Program II

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.

Control group--children who do not have reading problems

These are children with reading disabilities (without NF - ideopathic reading disabilities; IRD) who received Tutoring Program I

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.

These are children with reading disabilities (without NF - ideopathic reading disabilities; IRD) who received Tutoring Program II

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.

These are children who have reading disabilities (without NF) but did not receive intervention until after the trial.
Period Title: Overall Study
Started 8 9 26 14 18 14
Completed 8 9 26 14 18 14
Not Completed 0 0 0 0 0 0
Arm/Group Title NF-Tutoring Program 1 NF-Tutoring Program 2 Typically Developing Readers IRD- Tutoring Program 1 IRD-Tutoring Program 2 Waitlist Control Total
Hide Arm/Group Description

Tutoring Program I

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.

Tutoring Program II

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.

Control group--children who do not have reading problems

These are children with reading disabilities (without NF - ideopathic reading disabilities; IRD) who received Tutoring Program I

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.

These are children with reading disabilities (without NF - ideopathic reading disabilities; IRD) who received Tutoring Program II

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.

These are children who have reading disabilities (without NF) but did not receive intervention until after the trial. Total of all reporting groups
Overall Number of Baseline Participants 8 9 26 14 18 14 89
Hide Baseline Analysis Population Description
Participants both with and without NF1 were determined by researcher-administered assessments as having Reading Difficulties or typically developing reading skills.
Age, Continuous  
Mean (Standard Deviation)
Unit of measure:  Years
Number Analyzed 8 participants 9 participants 26 participants 14 participants 18 participants 14 participants 89 participants
9.62  (1.13) 11.06  (1.48) 9.70  (1.53) 10.08  (1.63) 10.33  (2.12) 10.72  (2.25) 10.18  (1.79)
Sex: Female, Male  
Measure Type: Count of Participants
Unit of measure:  Participants
Number Analyzed 8 participants 9 participants 26 participants 14 participants 18 participants 14 participants 89 participants
Female
5
  62.5%
1
  11.1%
13
  50.0%
6
  42.9%
9
  50.0%
3
  21.4%
37
  41.6%
Male
3
  37.5%
8
  88.9%
13
  50.0%
8
  57.1%
9
  50.0%
11
  78.6%
52
  58.4%
Woodcock Johnson Achievement Tests   [1] 
Mean (Standard Deviation)
Unit of measure:  Units on a scale
Number Analyzed 8 participants 9 participants 26 participants 14 participants 18 participants 14 participants 89 participants
78.25  (9.05) 85.78  (7.24) 103.81  (7.41) 77.57  (13.47) 79.67  (9.59) 79.64  (9.66) 86.88  (14.48)
[1]
Measure Description: This metric measures reading abilities (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 age-normed standard scores, with higher scores reflecting better performance on the measure (mean=100, standard deviation=15, such that a score >130 would be very superior and a score <70 would be very low). The Basic Reading score is a normed composite of the WJ-III subtests Letter-Word Identification and Word Attack, representing word-level reading skill.
1.Primary Outcome
Title Change From Baseline in WJ-III Basic Reading Normative Update (Woodcock Johnson Psychoeducational Battery – 3rd Edition; WJ-III NU) Standard Score at 15 Hours
Hide Description 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.
Time Frame 0 and 15 hours
Show Outcome Measure DataHide Outcome Measure Data
Hide Analysis Population Description
[Not Specified]
Arm/Group Title NF-Tutoring Program 1 NF-Tutoring Program 2 Typically Developing Readers IRD- Tutoring Program 1 IRD-Tutoring Program 2 Waitlist Control
Hide Arm/Group Description:

Tutoring Program I

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.

Tutoring Program II

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.

Control group--children who do not have reading problems

These are children with reading disabilities (without NF - ideopathic reading disabilities; IRD) who received Tutoring Program I

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.

These are children with reading disabilities (without NF - ideopathic reading disabilities; IRD) who received Tutoring Program II

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.

These are children who have reading disabilities (without NF) but did not receive intervention until after the trial.
Overall Number of Participants Analyzed 8 9 26 14 18 14
Mean (Standard Deviation)
Unit of Measure: units on a scale
13.50  (13.61) -1.11  (6.94) -.115  (5.13) 5.86  (9.31) 3.06  (9.93) .143  (7.97)
2.Secondary Outcome
Title Neuroimaging Data
Hide Description Neuroimaging data consists of functional MRI and structural MRI measures.
Time Frame Collected before and after intervention
Outcome Measure Data Not Reported
Time Frame [Not Specified]
Adverse Event Reporting Description [Not Specified]
 
Arm/Group Title NF-Tutoring Program 1 NF-Tutoring Program 2 Typically Developing Readers IRD-Tutoring Program 1 IRD-Tutoring Program 2 Waitlist Control
Hide Arm/Group Description

Tutoring Program I

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.

Tutoring Program II

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.

Control group--children who do not have reading problems These are children with reading disabilities (without NF - ideopathic reading disabilities; IRD) who received Tutoring Program I 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. These are children with reading disabilities (without NF - ideopathic reading disabilities; IRD) who received Tutoring Program II 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. These are children who have reading disabilities (without NF) but did not receive intervention until after the trial.
All-Cause Mortality
NF-Tutoring Program 1 NF-Tutoring Program 2 Typically Developing Readers IRD-Tutoring Program 1 IRD-Tutoring Program 2 Waitlist Control
Affected / at Risk (%) Affected / at Risk (%) Affected / at Risk (%) Affected / at Risk (%) Affected / at Risk (%) Affected / at Risk (%)
Total   --/--   --/--   --/--   --/--   --/--   --/-- 
Show Serious Adverse Events Hide Serious Adverse Events
NF-Tutoring Program 1 NF-Tutoring Program 2 Typically Developing Readers IRD-Tutoring Program 1 IRD-Tutoring Program 2 Waitlist Control
Affected / at Risk (%) Affected / at Risk (%) Affected / at Risk (%) Affected / at Risk (%) Affected / at Risk (%) Affected / at Risk (%)
Total   0/8 (0.00%)   0/9 (0.00%)   0/26 (0.00%)   0/14 (0.00%)   0/18 (0.00%)   0/14 (0.00%) 
Show Other (Not Including Serious) Adverse Events Hide Other (Not Including Serious) Adverse Events
Frequency Threshold for Reporting Other Adverse Events 0%
NF-Tutoring Program 1 NF-Tutoring Program 2 Typically Developing Readers IRD-Tutoring Program 1 IRD-Tutoring Program 2 Waitlist Control
Affected / at Risk (%) Affected / at Risk (%) Affected / at Risk (%) Affected / at Risk (%) Affected / at Risk (%) Affected / at Risk (%)
Total   0/8 (0.00%)   0/9 (0.00%)   0/26 (0.00%)   0/14 (0.00%)   0/18 (0.00%)   0/14 (0.00%) 
Small sample size; Different group sizes; Random assignment to treatment group (along with small sample size) produced some asymmetry of pretest score by treatment group, such that the NF-Tutoring Program 2 started with a higher group mean score.
Certain Agreements
Principal Investigators are NOT employed by the organization sponsoring the study.
There is NOT an agreement between Principal Investigators and the Sponsor (or its agents) that restricts the PI's rights to discuss or publish trial results after the trial is completed.
Results Point of Contact
Name/Title: Dr. Laurie Cutting
Organization: Vanderbilt University
Phone: 615-875-1054
Responsible Party: Laura Cutting, Vanderbilt University
ClinicalTrials.gov Identifier: NCT00624234     History of Changes
Other Study ID Numbers: R01NS049096 ( U.S. NIH Grant/Contract )
NF1-R01
R01NS049096 ( U.S. NIH Grant/Contract )
First Submitted: February 25, 2008
First Posted: February 27, 2008
Results First Submitted: August 5, 2016
Results First Posted: April 10, 2017
Last Update Posted: November 8, 2018