Abstracts
1. Tourette Syndrome: A Developmental
Psychobiologic View
Abstract
We
present a developmental psychobiologic framework to improve
the understanding of developmental and mental disorders. Tourette
syndrome (TS) is our model condition for the effort, as we review
our studies on the condition in conjunction with the comorbidities
with which it often presents. TS serves not only as a biologic
marker for certain localizable and definable central nervous
system processes, but also may serve a transacting function
across biological and psychological levels of organization.
Kerbeshian,
J. & Burd, L. Tourette Syndrome: A Developmental Psychobiologic
View. Journal of Physical and Developmental Disabilities 1994,
6, 203-218.
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2. A Educational Management of Children
With Tourette Syndrome
Abstract
In
a recent study of children with Tourette Syndrome (TS) in North
Dakota, 51% were found to have a learning disability. Fifty-seven
children from this study were found to have 1 or more other
developmental disabilities including mental retardation (26);
severe sensory impairment, blindness (9), deafness (3); nonspeaking
(10); severe school phobia (8); bipolar disorder (2); or schizophrenia
(3). We suggest that TS may be associated with other neuropsychiatric
developmental disorders more frequently than previously thought.
In 40% of the children with TS in North Dakota 1 or more developmental
disorders other than a learning disability, attention deficit
hyperactivity disorder or obsessive compulsive disorder co-occurs
with TS. This comorbidity contributes to severe and long lasting
social, emotional and educational problems for these children,
their families and schools. Diagnostic and management strategies
for educational management teams are needed to assist in the
development of appropriate intervention programs for these children.
More research to is necessary accomplish these goals.
Kerbeshian,
J. & Burd, L. Tourette Syndrome: A Developmental Psychobiologic
View. Journal of Physical and Developmental Disabilities 1994,
6, 203-218.
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3. Comorbid Tourette Syndrome and Bipolar
Disorder: An Etiologic Perspective
Abstract
We
attempt to elucidate relationships between Tourette Syndrome
(TS) and bipolar disorder (BD), using an epidemiologic approach.
Method: Of 205 TS patients, in our North Dakota TS surveillance
project 15 are comorbid for BD. A subset of these TS patients
had been included in earlier population based prevalence studies
of TS in children and adolescents, and adults. Minimal risk
ratios are calculated for TS+BD patients by age group (child
and adolescent or adult). This information is used to estimate
genetic risk indicators for TS and BD. Results: The risk of
developing BD in a population of children and adolescents or
adults with TS is increased more than 4 times above the level
expected by chance, but statistical significance is not achieved.
Conclusions: Comorbidity between TS and BD does not appear to
be due to chance co-occurrence of the two disorders. Although
a genetic mechanism may be causal, in the absence of family
studies, we offer an explanatory model.
Kerbeshian,
J. & Burd, L. Common Comorbidity Among Tourette's Syndrome,
Autistic Disorder and Bipolar Disorder. Journal of the American
Academy of Child and Adolescent Psychiatry 1996, 35, 681-685.
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4. A Family History Study of Comorbid
Tourette Syndrome and Bipolar Disorder
Abstract
Using
a family history approach, we attempt to generate hypotheses
regarding genetic and/or neurodevelopmental factors in a group
of patients comorbid for Tourette syndrome (TS) and bipolar
disorder (BD). Method: We identified 15 children and adolescents,
and adults with comorbid TS+BD. Using the same patient registry,
we then identified a comparison group of patients with garden
variety TS. We first reviewed charts of index patients and of
the comparison group using DSM-III-R criteria for TS and for
BD. We then reviewed charts of both groups for family histories
of tics, obsessive compulsive symptoms (OCS), and bipolar symptomatology
(BDS). Results: Our child and adolescent TS+BD group had a lower
than expected family history of tics and/or OCS and a greater
than expected family history of BDS. Our adult TS+BD group had
family histories of tics, OCS and BDS at the expected base rates.
Our adult TS+BD group had increased proportions of serious other
developmental comorbidities.
Discussion:
Our methodology allows for hypothesis generation, but does not
allow for hypothesis testing. This study identifies two separate
hypotheses that require further study: (1) In our child and
adolescent TS+BD group, do etiologic factors responsible for
BD lower the threshold for and advance the penetrance of genetic
factors for TS which otherwise might not have been expressed?
(2) In our adult TS+BD group with other serious comorbidities,
is the presence of BD due to genetic or neurophysiologic overlap
with TS and other comorbidities, or does the presence of TS
and other comorbidities enhance a low genetic risk for BD?
Kerbe
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5. Canalization as applied to Tourette
syndrome
Abstract
Tourette
syndrome (TS) is a developmental neuropsychiatric disorder,
the hallmark of which is the presence of multiple motor and
vocal tics with a duration of at least a year, and a childhood
or adolescent onset. TS is often comorbid with obsessive compulsive
disorder (OCD), which is felt to be an alternate expression
of a common genetic etiology for the condition. Individuals
with TS also likely to have a greater than chance concordance
with attention deficit-hyperactivity disorder (ADHD), pervasive
developmental disorder (PDD), and bipolar disorder (BD) (Kerbeshian
& Burd, 1992; Kerbeshian et al., 1995). A variety of genetic
models have been applied to explain the patterns of familiality
in TS and its comorbidities. These have included classical and
modified Mendelian models, polygenic models with multiple genes
of varying influence, and modifier gene models (Belmaker, 1991).
In spite of sophisticated and valiant efforts, a gene(s) for
TS has yet to be discovered. TS is likely a heterogeneous condition.
How then to conceptualize the emergence of TS and its comorbidities,
particularly in the absence of a reliable biological marker
and in the absence of explanatory family studies? We have found
the concept of canalization to be clinically and heuristically
valuable in this regard.
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6. Tourette Syndrome, Autistic Disorder
and Bipolar Disorder: Common Comorbidity and Its Implications
Abstract
Several
studies report a greater than expected concurrence for Tourette
syndrome (TS) with autistic disorder (AD). TS and bipolar disorder
(BD) also may co-occur at a greater than expected rate. In this
report we assess whether there is a greater than expected concurrence
for TS+AD+BD. Method: We identified 4 patients who had at some
time in their lives diagnoses of TS, AD, and BD. Three of these
had concurrent TS+AD+BD. Diagnoses were made utilizing DSM-III-R
criteria. Each of these patients was living in North Dakota
and was in our care at the time of this study. Results: The
point prevalence (risk) for concurrent TS+AD+BD in North Dakota
is not less than 4.6 x 10-6. The developmental sequence of syndromes
in these 4 patients was AD, then TS and then BD. Using data
from our previously published population based prevalence studies
we find that TS+AD+BD co-occur at a greater than chance expectation.
Conclusions Common etiologic factors may be involved in the
greater than chance concurrence of TS+AD+BD.
Kerbeshian,
J. & Burd, L. Common Comorbidity Among Tourette's Syndrome,
Autistic Disorder and Bipolar Disorder. Journal of the American
Academy of Child and Adolescent Psychiatry 1996, 35, 681-685.
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7. Neuropathology and Molecular Studies
of a Patient With Tourette Syndrome and Huntington Disease
Abstract
This
paper reviews the association of Huntington's Disease (HD) and
tic disorder or Tourette Syndrome (TS). We also present the
results of a followup study of a male with childhood-onset TS
and adult-onset HD who died at 45 years of age. The developmental
course and results of neuropathologic and molecular studies
of this patient are reported. This is the first case of childhood
onset of TS with adult onset of HD reported who has come to
autopsy. A developmental model for childhood and adult neuropsychiatric
disorders is presented.
Burd,
L., Kerbeshian, J., & Meredith, JL. Neuropathology and Molecular
Studies of a Patient with Tourette Syndrome and Huntington's
Disease. Journal of Developmental and Physical Disabilities
1997, 9 255-263.
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to titles
8. Tourette Syndrome: A Developmental
Psychobiologic View
Abstract
We
present a developmental psychobiologic framework to improve
the understanding of developmental and mental disorders. Tourette
syndrome (TS) is our model condition for the effort, as we review
our studies on the condition in conjunction with the comorbidities
with which it often presents. TS serves not only as a biologic
marker for certain localizable and definable central nervous
system processes, but also may serve a transacting function
across biological and psychological levels of organization.
Kerbeshian,
J., & Burd, L. Asperger's Syndrome and Tourette Syndrome:
The Case of the Pinball Wizard. British Journal of Psychiatry
1986, 148, 731-736.
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9. Tourette Syndrome and Learning Disabilities
Abstract
We
reviewed the records of 42 consecutive cases of children with
Tourette Syndrome (TS) who had IQs above 70 and contrasted the
reading, reading comprehension, math, and spelling quotients
with IQ scores to determine how many of these persons would
meet criteria for a learning disability. The mean IQ of the
35 males and 7 females was 94.4 and was higher than the mean
math score (82.8), spelling score (90.4), reading score (87.4)
and reading comprehension score of (85.3). Using a 1.5 standard
deviation discrepancy, 51% met criteria for learning disability
in at least one academic area; 21% had a 2 standard deviation
discrepancy. Children with TS are frequently learning disabled
and assessment of academic achievement should be a routine aspect
in the evaluation of such children.
Burd,
L., Kauffman, D. W. & Kerbeshian, J. Tourette Syndrome and
Learning Disabilities. Journal of Learning Disabilities 1992,
25, 598-604.
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10. Tourette Syndrome and Learning Disabilities
Abstract
A
review of the relationship between speech and language disorders
and tic disorders is presented. Selected aspects of neurophysiological
functioning linking speech and language disorders are reviewed.
A model for this relationship is presented. The utility of TS
as a model for understanding cognitive and linguistic functioning
is discussed.
Burd,
L., Kerbeshian, J., Leech, C. & Gascon, G. A Review of the
Relationship between Gilles de la Tourette Syndrome and Speech
and Language Disorder. Journal of Physical and Developmental
Disabilities 1991, 6(3), 1-19.
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11. Asperger's Syndrome and Tourette Syndrome:
The Case of the Pinball Wizard
Abstract
We
review the English-language literature on Asperger's syndrome
(AS), with particular reference to diagnostic criteria and differentiation
from infantile autism and personality disorders, and describe
six cases seen in practice: all met DSM-III criteria for atypical
pervasive developmental disorder'. Three also developed Tourette
syndrome: the co-occurrence of the two disorders, and methods
of intervention, are discussed.
Kerbeshian,
J., & Burd, L. Asperger's Syndrome and Tourette Syndrome:
The Case of the Pinball Wizard. British Journal of Psychiatry
1986, 148, 731-736.
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12. A Clinical Pharmacological Approach
to Treating Tourette Syndrome in Children and Adolescents
Abstract
Tourette
Syndrome ( TS) is a neuropsychiatric movement disorder characterized
by the presence of multiple motor and phonic tics. Monoamine
neurotransmitter dysfunction has been implicated in the expression
of the condition. Standard as well as novel pharmacologic treatments
for TS as a sole entity or as a condition co-morbid with attention
deficit-hyperactivity disorder (ADHD) and/or obsessive-compulsive
disorder (OCD) target these presumed neurotransmitter abnormalities.
Choice of a specific medication is also predicated upon an individual
patient's symptom profile, a cost-benefit analysis of desired
effects versus side effects, and the impact on co-morbid conditions.
Maximum involvement of the patient and parent or significant
other is encouraged. It is emphasized that pharmacologic treatment
is primarily symptomatic, usually not affecting the longer term
outcome of specific syndromes per se. The integration of pharmacologic
with psychoeducational interventions is encouraged.
Kerbeshian,
J. & Burd, L. A Clinical Pharmacological Approach to Treating
Tourette Syndrome in Children and Adolescents. Neuroscience
and Biobehavioral Reviews 1988, 12, 241-245
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13. Differential Responsiveness to Lithium
in Patients With Tourette Disorder
Abstract
The
current status of the use of lithium in the treatment of tic
disorders is reviewed. Areas of pathophysiologic overlap between
bipolar and tic symptomatology are explored from the standpoint
of shared etiology versus co-morbidity. Data from ten cases
of children and adolescents with tic disorders treated with
lithium are presented. Five exhibited a positive response to
tic and associated symptoms, while five did not. Treatment responders
versus nonresponders were compared along a number of parameters
including co-morbidity with other syndromes, family history,
prior medication history, medication used concurrently with
lithium, and medication used subsequent to treatment with lithium.
Differences between the two groups are explored.
Kerbeshian,
J. & Burd, L. Differential Responsiveness to Lithium in
patients with Tourette disorder. Neuroscience and Biobehavioral
Reviews 1988, 12, 247-250
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14. Anticonvulsant Medications: An Iatrogenic
Cause of Tic Disorders
Abstract
A
review of the relationship between anticonvulsant medications
and tics is presented. Data on 5 patients in whom anticonvulsants,
either caused tics or exacerbated existing tic disorders is
discussed. Discontinuation of the medication resulted in a decrease
in the frequency of tickings in all patients. The effects of
anticonvulsants on the reticular system are discussed. It is
felt that it may be important for clinicians to consider carefully
the use of barbiturate anticonvulsants, especially phenobarbital
in children with tics or a family history of tics. Tic disorders
caused or exacerbated by exposure to anticonvulsant medications
appear to be more common than previously reported, and in some
patients the tics may not remit with discontinuation of the
medication.
Burd,
L., Williams, M., Kjelstad, K., Schimke, A., & Kerbeshian,
J. Prevalence of Psychotropic and Anticonvulsant Drug Use Among
North Dakota Group Home Residents. Journal of Intellectual Disability
Research 1997, 41, 488-494.
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15. Tourette Disorder in North Dakota
Abstract
This
paper describes the symptom profiles of a large group of patients
with Gilles de la Tourette disorder (TD). In Part one we report
the results of a questionnaire study of patients in North Dakota
with TD. The data suggest that TD patients have high rates of
academic and social problems. Obsessive-compulsive symptomatology
is quite high in this population. This report is unique in that
these results are reported using an available data base to allow
for pooling of future research in TD patients in other settings.
In Part two of this paper we discuss important findings from
our clinic population of 130 TD patients.
Burd,
L., Kerbeshian, J., Cook, J., Bornhoeft, D. M. & Fisher,
W. Tourette Disorder in North Dakota. Neuroscience and Biobehavioral
Reviews 1988, 12, 223-228.
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16. Is Development of Tourette Disorder
a Marker for Improvement in Patients with Autism and Other Pervasive
Developmental Disorders?
Abstract
Fifty-nine
patients were identified whose early history was consistent
with infantile autism or other pervasive developmental disorders
(PDD). Twelve of these patients, later in their history, developed
symptoms consistent with Tourette disorder (TD). The group of
patients who developed TD scored significantly higher on measures
of IQ and receptive and expressive language. It is suggested
that the development of the symptoms of TD subsequent to the
onset of PDD may serve as a marker for improved developmental
outcome and that children with PDD who also meet criteria for
TD may constitute a distinct subgroup of children with PDD.
Implications for diagnosis and management are discussed.
Burd,
L. Fisher, W., Kerbeshian, J. & Arnold, M.E. Is Development
of Tourette Disorder a Marker for Improvement in Patients with
Autism and Other Pervasive Developmental Disorders? Journal
of the American Academy of Child and Adolescent Psychiatry 1987,
26 162-165.
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17. Tourette Disorder and Bipolar Symptomatology
in Childhood and Adolescence
Abstract
Three
boys with an early history of attention deficit disorder with
hyperactivity developed Tourette disorder. At 13, 12 and eight
years of age, respectively, each met DSM-III criteria for a
manic episode or bipolar disorder. Each of the boys had a family
history of affective or affective spectrum disorder. Lithium
carbonate in a range of 0.8 to 1.2 meq/L markedly improved their
bipolar symptomatology with Tourette symptoms improving in two
patients. Further study is suggested to determine the significance
of these findings.
Kerbeshian,
J. & Burd, L. Tourette Disorder and Bipolar Symptomatology
in Childhood and Adolescence. Canadian Journal of Psychiatry
1989, 34, 30-233.
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18. Letter: Symptom Substitution in Tourette
Disorder
Burd,
L., Kerbeshian, J. Symptom Substitution in Tourette Disorder.
Lancet, II, 1988, 1072.
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19. Letter: Treatment-generated Side Effects
from the use of Behavior Modification in Tourette Syndrome
Burd,
L. & Kerbeshian, J. Treatment Generated Side Effects from
the use of Behavior Modification in Tourette Syndrome. Developmental
Medicine and Child Neurology 1987, 29, 832-833.
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20. Diagnosis and management of Gilles
de la Tourette Disorder in rural North Dakota children
Abstract
Tourette
Disorder (TD) is more common than previously suspected. In the
state of North Dakota, the prevalence rate among school-aged
children is 5.2 per 10,000. In this paper the authors discuss
diagnosis and management of children with TD in a rural state.
Particular emphasis will be paid to identifying children for
referral from a public school or institutional setting, identifying
appropriate referral sources, and discussing methods to facilitate
multidisciplinary long-term management of these children.
Burd,
L. & Kerbeshian, J. Diagnosis and Management of Gilles de
la Tourette Disorder in Rural North Dakota Children. Rural Special
Education Quarterly 1988, 9, 20-26.
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21. Tourette Syndrome, Atypical Pervasive
Developmental Disorder and Ganser Syndrome in a 15-Year-Old, Visually
Impaired, Mentally Retarded Boy
Abstract
This is a case report of a 15-year-old visually impaired, mentally
retarded male who presents with symptoms consistent with Tourette
Syndrome, a Syndrome of approximate answers (Ganser's Syndrome)
and Atypical pervasive Developmental Disorder.
The
authors feel that this follow-up on the case presented earlier
by Parraga and Butterfield raises the possibility of a link
between a number of the symptoms of adult schizophrenia, appearing
in attenuated form in these two cases, and Tourette Syndrome.
Burd,
L., & Kerbeshian, J. Tourette Syndrome, Atypical Pervasive
Developmental Disorder, and Ganser Syndrome in a 15-year-old,
Visually Impaired, Mentally Retarded Boy. Canadian Journal of
Psychiatry 1985, 30, 74-76.
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22. Tourette Disorder and Schizophrenia
in Children
Abstract
In
North Dakota children, the prevalence rate for DSM-III schizophrenia
is 0.19 per 10,000 for both sexes; for males 0.35 per 10,000
and 0 per 10,000 for females. In this report we utilize case
studies to convey the symptomatic courses of the 2 childr patients
with DSM-III-R defined schizophrenia. Both patients first developed
Tourette Disorder (TD) and later developed schizophrenia by
DSM-III and by DSM-III-R criteria. Among North Dakota children
with TD the prevalence rate of schizophrenia is 8.7% for boys.
The ramifications of concordance for the two disorders are explored.
Kerbeshian,
J. & Burd, L. Tourette Disorder and Schizophrenia in Children.
Neuroscience and Biobehavioral Reviews 1988, 12, 267-270.
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23. Letter to Editor: Stuttering and Stimulants
Burd,
L., Kerbeshian, J. Stuttering and Stimulants. Journal of Clinical
Psycho-pharmacology 1991, 11, 72-73.
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24. Onset of Gilles de la Tourette's Syndrome
Before 1 Year of Age
Abstract
The
authors report on three patients in North Dakota with an apparent
onset of Gilles de la Tourette's syndrome before 1 year of age.
Infantile onset may occur in 4.1% of the child patients with
Tourette's disorder in that state. It is suggested that the
diagnostic criteria for Tourette's disorder be revised to include
patients who develop the illness before they are 1 year old.
Burd,
L. & Kerbeshian, J. Onset of Gilles de la Tourette Syndrome
Prior to One Year of Age. American Journal of Psychiatry 1987,
144, 1066-1067.
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25. Letter to Editor: Novel Side Effects
of Clonidine in the Treatment of Tourette Disorder
Kerbeshian,
J. & Burd, L. Novel Side Effects of Clonodine in the Treatment
of Tourette Disorder. Journal of Clinical Psychopharmacology
1987, 7 207-208.
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25. Letter to Editor: Novel Side Effects
of Clonidine in the Treatment of Tourette Disorder
Kerbeshian,
J. & Burd, L. Novel Side Effects of Clonodine in the Treatment
of Tourette Disorder. Journal of Clinical Psychopharmacology
1987, 7 207-208.
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26. Familial Pervasive Development Disorder,
Tourette Disorder and Hyperlexia
Abstract
Four
children and one adult have been found to have pervasive Developmental
Disorder, Tourette disorder and hyperlexia in North Dakota,
a state with a population of 204,161 children ages ?18. Assuming
that these are independent disorders the probability of these
three disorders occurring by chance in one child is 3.39x10-12.
Two of these individuals are from the same family. This suggests
evidence for genetic linkage among these three disorders.
Kerbeshian,
J. & Burd, L. Familial Pervasive Development Disorder, Tourette
Disorder and Hyperlexia. Neuroscience and Biobehavioral Reviews
1988, 12, 233-234.
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27. Letter to the Editor: A Case of Autism
and Mosaic of Trisomy 8
Abstract
A
family with fragile-X syndrome is reported. One sibling has
atypical pervasive developmental disorder and moderate mental
retardation. A second sibling has Tourette's syndrome, moderate
mental retardation. A second sibling has Tourette's syndrome,
moderate mental retardation, seizure disorder, and autism. A
third sibling has attention deficit disorder, moderate mental
retardation, and developmental language disorder, expressive
type. The authors believe that this family represents a classic
example of the differential outcome of interactions of common
biogenetic and environmental influences. We propose that in
this family the multipotential outcome is at least influenced
by if not caused by a common genetic defect.
Kerbeshian,
J., Burd, L., & Martsolf, J. Brief Communication: A Family
with Fragile X Syndrome. The Journal of Nervous and Mental Disease
1984, 172, 549-551.
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28. Fragile X Syndrome Associated with
Tourette Symptomatology in a Male with Moderate Mental Retardation
and Autism
Abstract
A
case of fragile X syndrome and Tourette symptomatology in an
11-year-old mentally retarded male with autism is reported.
The coexistence of these multiple disorders has apparently not
been previously reported.
Kerbeshian,
J., Burd, L., & Martsolf, J. Brief Report: Fragile X Syndrome
Associated with Tourette Symptomatology in Male with Moderate
Mental Retardation and Autism. Journal of Developmental Behavioral
Pediatrics 1984, 5, 201-203.
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29. Letter to Editor: Nocturnal Coprolalia
and Phonic Tics
Burd,
L. & Kerbeshian, J. Nocturnal Coprolalia and Phonic Tics.
American Journal of Psychiatry 1988, 145, 132.
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30. Pseudohemiparesis and Tourette Syndrome
Abstract
Three
patients with Tourette syndrome and transient recurrent hemiparetic
posturing (pseudohemiparesis) are presented. The transient nature
of this posturing is not consistent with a static central nervous
system deficit. It is felt that the Tourette syndrome symptomatology
and pseudohemiparesis share a common pathophysiology. The inclusion
of pseudohemiparesis in the differential diagnosis for cerebral
palsy is suggested.
Burd,
L., Kerbeshian, J., Fisher, W., Barcome, D. & Lipp, L. Pseudohemiparesis
and Tourette Syndrome. Journal of Child Neurology 1986, 1, 369-371.
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31. Tourette Disorder and Mutational Falsetto
Abstract
Speech,
language and voice disorders associated with Tourette Disorder
(TD) are reviewed. Two cases of TD associated with falsetto
are presented. The relationship between abnormalities in vocal
pitch and tic symptomatology is explored.
Kerbeshian,
J. & Burd, L. Tourette Disorder and Mutational Falsetto.
Neuroscience and Biobehavioral Reviews 1988, 12, 271-273.
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32. A Comparison of the Effects of Parental Risk Markers on Pre and Perinatal Variables in Multiple Patient Cohorts with Fetal Alcohol Syndrome, Autism, Tourette and Sudden Infant Death Syndrome: An Enviromic Analysis
Abstract
The prevalence and magnitude of effect of individual risk markers for specific developmental varies widely across diagnostic category. The four study cohorts for this project were patients from four diagnostic registries in North Dakota for fetal alcohol syndrome, autism, sudden infant death syndrome (SIDS), and tourette syndrome. These four cohorts were used to estimate prevalence and magnitude of effect of parental risk markers in patients with developmental disabilities. Cases with North Dakota birth certificates were matched with controls. Using birth certificate data, we then examined five parental risk markers for each cohort and estimated direct and indirect effects for each risk marker by cohort. The authors found two significant paternal risk markers (age in SIDS and education in fetal alcohol syndrome.) Significant maternal markers were (age in SIDS, education in fetal alcohol syndrome, autism and SIDS). Marital status was a significant risk marker in fetal alcohol syndrome. Effect size using paired t-tests, odds ratios, and population attributable risk for both direct and indirect effects for each marker. We estimated to allow for direct comparisons of the differential effect estimates of each of these markers. The direct effect of parental markers differs across diagnostic cohorts of patients. Use of cohorts from similar denominator populations primarily obtained from prevalence studies of the cohort is a useful methodologic tool for estimating the prevalence and magnitude of effect of risk markers
Klug, M.G., Burd, L., Kerbeshian, J., Benz, B., & Martsolf, J.T. A Comparison of the Effects of Parental Risk Markers on Pre and Perinatal Variables in Multiple Patient Cohorts with Fetal Alcohol Syndrome, Autism, Tourette Syndrome, and Sudden Infant Death Syndrome: An Enviromic Analysis. Neurotoxicology and Teratology (in press).
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33. Long-term follow-up of an epidemiologically defined cohort of patients with Tourette syndrome.
Abstract
The goal of this study was to collect prospective longitudinal information on the development of an epidemiologically defined cohort of patients with Tourette syndrome. These data may improve prognostic understanding of the condition. This information will also be important for specification of an adult phenotype for genetic marker studies. A prospective longitudinal cohort study was conducted. Fifty-four of 73 patients from our 1986 prevalence study of Tourette syndrome in North Dakota school-aged children were eligible for inclusion. The subjects were diagnosed in 1984 and 1985. We were able to interview 39 of 54 eligible patients for 507 person-years of follow-up. For the cohort, tic severity declined by 59%, global assessment of functioning improved by 50%, and the average number of comorbidities decreased by 42%. Forty-four percent of patients were essentially symptom free at follow-up. Only 22% were on medication as adults. Tourette syndrome is a developmental neuropsychiatric disorder with a long-term course that is favorable for most patients. Males demonstrated substantially more variability in improvement but overall demonstrated more improvement than females.
Burd, L., Kerbeshian, J., Barth, A., Klug, M., Avery, K., & Benz, B. Long-Term Follow-up of an Epidemiologically Defined Cohort of Patients with Tourette Syndrome. Journal of Child Neurology 2001, 16(6), 431-437.
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34. An International Perspective on Tourette Syndrome: Selected Findings from 3,500 Individuals in 22 Countries.
Abstract
We have established a multisite, international database of 3,500 individuals diagnosed with Tourette syndrome (TS). The male:female ratio is 4.3:1 for the total sample, with wide variation among sites; the male excess occurs at every site. Anger control problems, sleep difficulties, coprolalia, and self-injurious behavior only reach impressive levels in individuals with comorbidity. Anger control problems are strongly correlated with comorbidity, regardless of site, region, or whether assessed by neurologists or psychiatrists. The mean age at onset of tics is 6.4 years. At all ages, about 12% of individuals with TS have no reported comorbidity. The most common reported comorbidity is attention-deficit-hyperactivity disorder. Males are more likely to have comorbid disorders than females. The earlier the age at onset, the greater the likelihood of a positive family history of tics. An understanding of the factors producing these and other variations might assist in better subtyping of TS. Because behavioral problems are associated with comorbidity, their presence should dictate a high index of suspicion of the latter, whose treatment may be at least as important as tic reduction. The established database can be used as the entry point for further research when large samples are studied and generalizability of results is important.
Freeman, R.D., Fast, D.R., Burd, L., Kerbeshian, J., & Robinson, M.M. An International Perspective on Tourette Syndrome: Selected Findings from 3,500 Cases in 22 Countries. Developmental Medicine and Child Neurology 2000, 42(7), 436-447
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35.Comorbid Down's Syndrome, Tourette Syndrome and Intellectual Disability: Registry Prevalence and Developmental Course
Abstract
The co-occurrence of Tourette syndrome (TS) and Down's syndrome (DS) has been previously reported in the literature. In the present study, a retrospective record review was conducted using the North Dakota TS registry in order to ascertain the number of cases of TS and DS, and to develop case descriptions. We identified five cases from North Dakota. Two of these patients were simply comorbid for TS and DS. One was additionally comorbid for bipolar disorder, another for childhood disintegrative disorder and a third had a D/G group translocation. The association between DS and TS occured in 2% of TS patients. Contrary to the situation in patients with pervasive developmental disorders, the presence of TS in DS may be a negative prognostic indicator.
Kerbeshian, J., Burd, L. Comorbid Down’s Syndrome, Tourette Syndrome and Intellectual Disability: Registry Prevalence and Developmental Course. Journal of Intellectual Disability Research 2000, 44, 60-67.
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36.Peek-a-boo Fragile Site at 16d Associated with Tourette Syndrome, Bipolar Disorder, Autistic Disorder, and Mental Retardation.
Abstract
Five patients with a fragile site at 16q22-23 and neuropsychiatric disorders are reported. Three of five had Tourette disorder, three had mental retardation, two had bipolar disorder, and one had autistic disorder. During our attempts to study the fragile sites in more detail we were unable to reproduce the fragile sites found several years earlier. The potential relationship between the fragile sites and the neuropsychiatric disorders in these patients is discussed. Am. J. Med. Genet. (Neuropsychiatr. Genet.) 96:69-73, 2000. Copyright 2000 Wiley-Liss, Inc.
Kerbeshian, J., Severud, R., Burd, L., & Larson, L. A Peek-A-Boo Fragile Site at 16D Associated with Tourette Syndrome, Bipolar Disorder, Autistic Disorder, and Mental Retardation. American Journal of Medical Genetics 2000, 96(1), 69-73.
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37. Recognition and Management of Tourette's Syndrome and Tic Disorders.
Abstract
Tic disorders and Tourette's syndrome are conditions that primary care physicians are likely to encounter. Up to 20 percent of children have at least a transient tic disorder at some point. Once believed to be rare, Tourette's syndrome is now known to be a more common disorder that represents the most complex and severe manifestation of the spectrum of tic disorders. Tourette's syndrome is a chronic familial disorder with a fluctuating course; the long-term outcome is generally favorable. Although the exact underlying pathology has yet to be determined, evidence indicates a disorder localized to the frontal-subcortical neural pathways. Tourette's syndrome is commonly associated with attention-deficit/hyperactivity disorder, obsessive-compulsive disorder, behavior problems and learning disabilities. These comorbid conditions make the management of Tourette's syndrome more challenging. Management of Tourette's syndrome should include timely and accurate diagnosis, education, and behavior or pharmacologic interventions. Use of neuroleptic medications and dopamine D2 antagonist drugs can be effective but may be associated with significant side effects.
Bagheri, M., Kerbeshian, J., & Burd, L. Recognition and Management of Tourette Syndrome and Tic Disorders in Primary Care. American Family Physician 1999, 59, 2263-2272.
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38. Prenatal and Perinatal Risk Factors for Tourette Disorder.
Abstract
AIMS: To identify pre- and perinatal risk factors for Tourette disorder.
METHODS: Case control study. We matched names of patients who met DSM criteria for Tourette disorder with their birth certificates. For each case five controls were selected. The controls were matched by sex, year and month of birth.
RESULTS: Univariate analysis of the 92 cases and the 460 matched controls identified 4 risk factors; one categorical variable--trimester prenatal care begun and 3 continuous variables--apgar score at 5 minutes, month prenatal began and number of prenatal visits. Logistic modeling to control for confounding produced a three variable model (apgar score at 5 minutes (OR = 1.31), number of prenatal visits (OR = .904) and fathers age (OR = .909). The model parameters were: chi 2 = 19.76; df = 3; p < .001.
CONCLUSIONS: This is an inexpensive methodology to identify potential risk factors of patients with Tourette disorder and other mental illness.
Burd, L., Severud, R., Klug, M.G., & Kerbeshian, J. Prenatal and Perinatal Risk Factors for Tourette Disorder. Journal of Perinatal Medicine 1999, 27, 295-302.
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39. Comorbid Tourette's Disorder and Bipolar Disorder: an Etiologic Perspective.
Abstract
OBJECTIVE: Using an epidemiologic approach, the authors attempt to elucidate relationships between Tourette's disorder and bipolar disorder.
METHOD: Of 205 patients with Tourette's disorder in the North Dakota Longitudinal Tourette Syndrome Surveillance Project, 15 had comorbid bipolar disorder. A subset of the patients with Tourette's disorder had been included in earlier population-based prevalence studies of Tourette's disorder in children, adolescents, and adults. Minimal risk ratios were calculated for the patients with Tourette's disorder plus bipolar disorder by age group (children/adolescents and adults). This information was used to estimate genetic risk indicators for comorbid Tourette's disorder and bipolar disorder.
RESULTS: The estimated risk of developing bipolar disorder among the study group of children, adolescents, and adults with Tourette's disorder was more than four times higher than the level expected by chance, but this finding did not reach statistical significance. It was indicative of trends, however.
CONCLUSIONS: Comorbidity between Tourette's disorder and bipolar disorder does not appear to be due to chance co-occurrence of the two disorders. Although a genetic mechanism may play a causal role, in the absence of family studies an explanatory model involving the concept of canalization of basal-ganglia-mediated dysfunctions is offered. In such a construct, Tourette's disorder would be a likely accompaniment to other conditions, including bipolar disorder, whose pathogenic determinants might channel through neural pathways involving the basal ganglia. The presence of significant developmental disabilities may further enhance factors culminating in comorbid Tourette's disorder and bipolar disorder.
Kerbeshian, J., Burd, L. Tourette’s Disorder and Bipolar Disorder: An Etiologic Relationship. American Journal of Psychiatry 1995, 151, 1646-1651.
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40. Huntington Disease and Childhood-Onset Tourette Syndrome.
Abstract
A 40-year-old man with childhood-onset Tourette syndrome (TS) developed Huntington disease (HD). We believe this to be the first reported case of childhood-onset TS with adult onset HD. Discovery of other cases with both disorders may provide clues to the pathophysiology of both conditions.
Kerbeshian, J., Burd, L., Leech, C., & Rorabaugh, A. Huntington Disease and Childhood-Onset Tourette Syndrome. American Journal of Medical Genetics 1991, 39, 1-3.
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41. Peek-A-Boo Fragile Site? Or a Peek-A-Boo Paper?
Abstract
Kerbeshian, J., Burd, L. Peek-A-Boo Fragile Site? Or a Peek-A-Boo Paper? American Journal of Medical Genetics 2000, 96(3), 430-431.
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42. A Role for Biogenic Amines in Developmental Language Disorders.
Abstract
The authors discuss the current uncertainty regarding etiology, treatment, and classification of developmental language disorders (DLD). Referring to previous reports in the literature, they propose a specific subclass of DLD associated with a hypothesized dysfunction of the dopaminergic system of the basal ganglia. Mechanisms of dysfunction and treatment implications are discussed.
Kerbeshian, J., Gascon, G., & Burd, L. A Role for Biogenic Amines in Developmental Language Disorders. Neuroscience and Biobehavioral Reviews 1988, 12, 289-294.
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43. Fragile X Syndrome: Developmental Aspects
Abstract
Burd, L., Bornhoeft, D.M., & Kerbeshian, J. Fragile X Syndrome: Developmental Aspects. Child Study Journal 1986, 16, 285-296.
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44. Anticonvulsant Medications: an Iatrogenic Cause of Tic Disorders.
Abstract
A review of the relationship between anticonvulsant medications and tics is presented. Data on 5 patients in whom anticonvulsants, either caused tics or exacerbated existing tic disorders is discussed. Discontinuation of the medication resulted in a decrease in the frequency of tickings in all patients. The effects of anticonvulsants on the reticular system are discussed. It is felt that it may be important for clinicians to consider carefully the use of barbiturate anticonvulsants, especially phenobarbital, in children with tics or a family history of tics. Tic disorders caused or exacerbated by exposure to anticonvulsant medications appear to be more common than previously reported, and in some patients the tics may not remit with discontinuation of the medication.
Burd, L., Kerbeshian, J., Fisher, W., & Gascon, G. Anticonvulsant Medications: An Iatrogenic Cause of Tic Disorders. Canadian Journal of Psychiatry 1986, 31(5), 419-423.
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45. Multiply Disabled Children
Abstract
Burd, L., Fisher, W. Multiply Disabled Children. Rehabilitation Literature 1985, 46, 243-245.
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46. Integrating Developmental, Pharmacologic and Psychologic Diagnosis and Management Through the Transdisciplinary Team Process
Abstract
Fisher, W., Burd, L., & Kerbeshian, J. Integrating Developmental, Pharmacologic and Psychologic Diagnosis and Management Through the Transdisciplinary Team Process. Rehabilitation Literature 1985, 46, 268-271
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