Cherab Foundation’s First Apraxia Conference
The First Scientific Conference Dedicated to the Therapy of Verbal Apraxia/Dyspraxia. Professional anecdotal overseen by CAPT Joseph Hibbeln, M.D.
The following was from ‘The First Apraxia Conference’ July 23-24, 2001, Headquarters Plaza Hotel, Morristown, New Jersey USA and was also presented at the Research Workshop – September 20-21 and on September 22, 2001 ‘Fatty Acids in Neurodevelopmental Disorders’ St Anne’s College, Oxford, UK
CHERAB FOUNDATION SCIENTIFIC PROGRAMS
The first scientific conference for therapy of verbal apraxia/dyspraxia entitled: “Verbal Apraxia/Dyspraxia and Essential Fatty Acid (EFA) Supplementation: A New Potential Therapeutic Intervention,” 23-24 July, 2001, Headquarters Plaza Hotel, Morristown, New Jersey, U.S.A., was organized under the auspices of the CHERAB Foundation and CAPT Joseph Hibbeln, M.D. The research findings described below were presented by CHERAB Foundation professional staff to a panel of participating experts for their review. The panel recommended the initiation of clinical trials to validate the potential therapeutic effects of EFA supplementation in verbal apraxia and autism. The data was also presented as three posters at the Conference on “Fatty Acids in Neurodevelopmental Disorders”, September 20-21, 2001 Oxford, United Kingdom.
VERBAL APRAXIA/DYSPRAXIA and the THERAPEUTIC ROLE of ESSENTIAL FATTY ACIDS:
Marilyn C. Agin, M.D., New York City Early Intervention, New York, and CHERAB Foundation, Gillette, New Jersey Robert Katz, Ph.D., Consortium for Fatty Acids, Omega-3 Research Institute, Inc., Bethesda, Maryland and CHERAB Foundation, Gillette, New Jersey Lori L. Roth, CCC SLP, CHERAB Foundation, Gillette, New Jersey Verbal Apraxia (VA) affects the programming of the articulators and rapid sequences of muscle movements for speech sounds. These children frequently display neurologic “soft signs” including hypotonia, sensory integration disorder, and motor planning difficulties. The speech assessment reveals a limited repertoire of consonant sounds, inconsistency of speech errors, and sound/syllable omissions. These children usually have near-normal receptive language and intelligence. It is a difficult speech disorder to treat with variable outcomes. Many children never develop intelligible, conversational speech. Dramatic leaps in speech progress have been noted with essential fatty acid (EFA) supplementation by parents and professionals. The most often used EFA supplement is a 1000 mg capsule containing a mixture of DHA (docosahexaenoic acid, 99 mg.), EPA (eicosapentaenoic acid, 148 mg.), GLA (gamma-linolenic acid, 40 mg) available under the name of ProEFA or Complete Omega and manufactured by Nordic Naturals, California.
The Perspectives of Speech Pathologists:
Our objective was to assess potential therapeutic effects of essential fatty acid (EFA) supplementation of children with VA by surveying speech pathologists that provide speech therapy to the supplemented children. A total of nineteen speech pathologists (eighteen of them independent), returned the questionnaires that constituted the professional anecdotal case reports included in this analysis. The patient population consisted of 16 males (including one pair of identical twins) and 3 females, mostly between 27-97 months of age. Seventeen of the 19 patients were supplemented with ProEFA (13 with one 1000 mg softgel capsule/day and four with two). Outcome variables measured included the following: speech, affect, muscle tone, muscle control, behavior, social skills, attention, eye contact, and academic ability. Post-supplementation, the children were rated according to the following scale: 1=not sure, 2=no change, 3=subtle change, 4=moderate 5=significant, 6=outstanding change. The analysis of data led to the following conclusions: a) EFA supplementation resulted in a marked shift in verbal statement ability from the nonverbal end toward the singing end of a hierarchical sequence, i.e., from decreases in nonverbal, gesturing, grunting, single sounds, to increases in single words, multiple words, sentences and singing. b) Seventeen of the 19 subjects (89%,) showed varied degrees of improvements in the Speech outcome variable. Of these improvements 9 (53%) were subtle, 5 (29%) were moderate and 3 (18%) were significant. Only two patients (11% of 19) showed no improvement. The nineteen reports were divided in two subgroups according to the effect of supplementation on the speech/communication outcome variable. A statistical test indicated that improvements in speech of patients in Subgroup 2 (containing all eight cases representing moderate and better than moderate improvements cores of 4 and 5 respectively), are significantly higher than the improvements in speech of patients in Subgroup 1 (containing the eleven cases representing the no-change and subtle improvement scores of 2 and 3 respectively). Three patients in Subgroup 2 were diagnosed with verbal apraxia (one mild, one moderate and one severe case). In addition, the mild case also had feeding-swallowing disorder. A fourth patient was diagnosed with mild VA and oro-motor hypotonia, additional three patients had severe VA with hypotonia and sensory integration disorder (SID). One of these also had autism and another was suspected to have ADHD. The eighths patient had mild VA with hypotonia and SID. The patient with autism showed moderate improvement in speech and better than moderate improvements in behavior and attention. Descriptive statistics (mean and standard deviation) of all variables surveyed in the population of Subgroup 2 indicate (in order of decreasing means) that improvements in Speech (4.4(0.5) > Attention (4.0(1.5) = Behavior (4.0(1.9) > Affect (3.4(0.6) = Social Skills (3.4 (1.4) = Eye Contact (3.4(1.7) > Muscle Tone (3.1(1.6) > Muscle Control/Coordination (2.7(1.0).
The Perspective of a Developmental Pediatrician:
Anecdotal case reports provided by the CHERAB Foundation’s Developmental Pediatrician were also analyzed. Ten children were supplemented: Nine had the diagnosis of VA; one had a dual diagnosis of VA and pervasive developmental disorder (PDD-NOS), and one was autistic with an expressive language disorder. Eight of the children were receiving 1 capsule/day of ProEFA and 2 were receiving 2 capsules/day. The majority of the children had been supplemented for at least three months. All of the children were receiving speech therapy at least three times a week. Age range was 32 months to 96 months old. Descriptive statistics were used to analyze the data. The same outcome variables and scoring scales have been used as above. The variables that showed the most improvement were speech and attention, with means of 4.7 (SD=1.3) and 4.1 (SD=1.2) respectively. According to the scale, this correlated with moderate to significant improvement. To a lesser degree, there was improvement in affect and eye contact with means of 3.8 for both. There were no significant changes in the other variables. The two children on the autistic spectrum showed significant improvements in speech and eye contact, with means of 5.0 and 5.5 respectively.
A Time Line of Therapeutic Outcomes in Speech/Communication
Speech therapy intervention has been an integral part of a program designed to treat children diagnosed with VA. Speech therapy approaches from oral motor patterning to “traditional” articulation drills yield fair success over lengthy periods of time. The potential therapeutic effect of EFA supplementation initiated by parents was followed in four children with VA by the CHERAB Foundation’s speech pathologist. Outcomes of the study are reported here. An initial evaluation consisting of a receptive and expressive language test, oral motor coordination examination and verbal/sound production test (Receptive One Word Vocabulary Test, Expressive One Word Vocabulary Test, Preschool Oral Motor Functioning Scale, Kaufman Speech Praxis Test) was performed on each subject prior to EFA supplementation. In general, the children demonstrated age-appropriate receptive language skills, extreme difficulty coordinating articulator movements for sound production, and a significant delay in expressive language skills. The children were given a daily dose of one 1000mg capsule of ProEFA. Two weeks into supplementation, each child began demonstrating improved attention to task, sustained eye contact with the therapist and calmer general participatory behavior. Beyond this time, each child demonstrated an improvement in the level of verbal statement specific to the baseline performance obtained in testing. One of the children began supplementation essentially non-verbal and progressed to two-word utterance production within 2 months. Outcome measures included standardized scores from general tests of language and measures taken from language-sample analyses as well as an objective scale grading speech production from non-verbal to singing. There were modest to significant changes in standardized measures of language after 2-3 months of EFA supplementation in all four cases using an 80% criterion confidence interval. These were substantiated by the clinically significant changes in language sample measures. Such improvement characteristically occurs after 9 to 12 months of intensive speech therapy intervention.
The above preliminary data provide evidence that: EFA supplementation has great potential in accelerating speech gains in children with verbal apraxia/dyspraxia. Thus, EFA supplementation in conjunction with speech therapy improved pre-speech behaviors (eye-contact, attention to task), speech and language production (single sound, word and sentence production), imitation skill accuracy and decreased inconsistent imitation errors, distractibility and groping behaviors.
Improvements are greater than would be expected from speech therapy alone
Verbal apraxia appears to be present in a percentage of children on the autistic spectrum and an association could be possible between VA and other disorders/syndromes, such as: hypotonia, sensory integration disorder, dysarthria, attention deficit hyperactivity disorder, Kabuki Syndrome and cerebral palsy. Further exploration of the basic and clinical aspects of these phenomena appears warranted.
A panel of scientific experts at the July 23-24 Conference discussed the evidence presented above and unanimously agreed that the existing scientific evidence justifies planning and implementing a comprehensive clinical trial to convincingly validate this new, potential therapeutic intervention. The panel discussed various clinical research alternatives and recommended that a randomized, placebo-controlled multi-center clinical trial of EFA and placebo supplementation to be undertaken as soon as possible. For example, all diagnosed verbal apraxic children, including those with hypotonia and sensory integration disorder, who have not been supplemented with EFAs, would be eligible for randomization. The panel suggested that all randomized children would be supplemented with EFA or placebo in addition to appropriate speech therapy. This took into consideration the potential cooperative or possibly synergistic effect of the combined therapies in the treatment group. The length of the trial is proposed to be 3 months. Improvement in verbal communication
skills, or the lack thereof, using an assessment protocol as described above, would be the major therapeutic outcome measured, and plasma, as well as erythrocyte membrane EFA levels would be monitored periodically. The two groups would consist of about 20-30 age-matched subjects. ProEFA would be the therapeutic supplement used in the trial based on preliminary successes attained.
Post Conference Statement
History How This Conference Came About
CHERAB FOUNDATION PROFESSIONAL STAFF
Marilyn C. Agin, M.D., Medical Director, CHERAB Foundation, graduated from New Jersey Medical School in 1986, followed by a combined residency in Pediatrics and Physical Medicine and Rehabilitation at New York University Medical Center. She is board certified in both fields. Prior to medical school, Dr. Agin received her master’s degree in Communication Disorders and was a practicing speech pathologist. Currently, Dr. Agin is the Medical Director of the New York City Early Intervention Program and does private neurodevelopmental evaluations primarily for children with communication disorders, learning disabilities, and autism,. She is a member of the New York City chapter of the Committee on Children with Disabilities of the American Academy of Pediatrics (AAP), and has been appointed to the Executive Council of the New York City chapter of the AAP.
Robert Katz, Ph.D., Director for EFA Research, CHERAB Foundation, received his degree in Organic/Medicinal Chemistry from the Hebrew University, Jerusalem in 1972. During his postdoctoral fellowship (1972-1973) at the National Institutes of Health (NIH), Bethesda, Maryland he worked in computer-assisted drug design of analgesics and molecular pharmacology of neurotransmitters. From 1978 to 1993, Dr. Katz was Director of Metabolic Diseases Research Program, National Institute of Diabetes, and Digestive and Kidney Diseases, NIH where he administered and managed nation-wide research programs in membrane structure and function, membrane protein crystallization, structural biology (proteomics), enzyme replacement and gene therapy, etc,. He organized workshops and conferences in these areas and identified research directions in need of development. Since leaving the NIH, (1993), Dr. Katz has developed DHA- and EPA-derivatized polycationic-lipophilic drug carriers to the CNS. In 1998 he founded the Omega-3 Research Institute, Inc. (O3RI), where he co-organized international workshops on omega-3 fatty acids in brain function, in diabetes and its cardiovascular complications, in molecular and cellular aspects of cancer and recently in verbal apraxia/dyspraxia.
During the last year Dr. Katz founded the Consortium for Brain Fatty Acids, O3RI, a “center without walls” that is providing a broad range of expert research support to parties that require such. Dr. Katz is co-developing the CHERAB Foundation’s EFA-based programs.
Lori L. Roth, MA, CCC-SLP, is a CHERAB Foundation Speech-Language Pathologist and Oral Motor Specialist with over 25 years of experience. She received her BA degree in Psychology from G. Washington University in Washington, D.C. in 1972. In 1974 she was awarded her Masters of Speech and Audiology from the Catholic University of America in Washington, DC. Ms. Roth’s experience includes home- and hospital-based rehabilitation, private and public school intervention and private practice. Lori Roth was instrumental in establishing an Infant Stimulation Program (called Early Intervention) in Annapolis, Maryland. She has mentored and trained graduate students in Speech and Language Pathology from New York University, Columbia University, Montclair State University and The College of New Jersey as well as practicing therapists in the State of New Jersey. Ms. Roth has presented professional workshops for colleagues and regularly acts as a consultant for private and public schools.
Organizers and Scientific Panel Members of the First Conference on Verbal Apraxia/Dyspraxia
“Verbal Apraxia/Dyspraxia and Essential Fatty Acid (EFA) Supplementation: A New Potential Therapeutic Intervention,” 23-24 July, 2001, Headquarters Plaza Hotel, Morristown, New Jersey, U.S.A.,
Marilyn C. Agin, M.D., Medical Director, Early Intervention, New York City, New York, and Medical Director, CHERAB Foundation, Gillette, New Jersey. (Also a scientific panel member)
Robert Katz, Ph.D., Managing Director, Consortium for Brain Fatty Acids (CFBFA), Omega-3 Research Institute, Inc., Bethesda, Maryland and EFA Director of Research, CHERAB Foundation, Gillette, New Jersey (Also a scientific panel member).
Scientific Panel Members:
Susan E. Carlson, Ph.D., Professor, University of Kansas, Kansas City, Kansas, Member Consortium for Brain Fatty Acids (CFBFA)
Joseph Hibbeln, M.D., Chief, Outpatient Clinic National Institute of Alcoholism and Alcohol Abuse, NIH, Bethesda, Maryland, Non-affilited Collaborator, CFBFA
Nancy Kaufman, M.A., CCC/SLP, Director, Kaufman Children’s Center for Speech Language and Sensory Disorders, West Bloomfield, Michigan
Ann Moser,B.S., Manager, Peroxisomal Diseases and Fatty Acid Profiles Clinical Laboratory,Kennedy Krieger Institute, Baltimore, Maryland. (Also a component laboratory of the CFBFA)
Jennifer Hill-Karrer, Ph.D., Associate Professor, University of Kansas Medical Centre, Kansas City, Kansas, and Collaborator CFBFA
Lori Roth M.A., CCC/SLP, Speech Pathologist, CHERAB Foundation
Andrew Zimmerman, M.D., Professor, Johns Hopkins University and Kennedy Krieger Institute, Baltimore, Maryland, and Collaborator, CFBFA.
Alexandra J. Richardson, MA, D.Phil., Senior Research Fellow in Neuroscience, Imperial College School of Medicine, MRI Unit, Hammersmith Hospital, London; and University Lab. of Physiology, Oxford.
The Administrative Organizers:
Lisa Geng, President
Suzanne Smolyar, Executive Vice President
Glenn W. Geng Executive Director, Treasurer