PEDIA TRIC R EHA BILITA TION ,
1, 29 ± 35
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For personal use only.
1999, VOL . 3, NO.
The prevalence of attentional problems and the
e€ ect of methylphenidate in children with
myelomenigocele
MICHAEL DAVIDOVITCH*, PATR ICIA MANNING-COURTNEY,
LINDA A. HARTMANN, JENNIFER WATSON,
MARLENE LUTKENHOFF and SONYA OPPENHEIMER
Accepted for publication: December 1998
Keywords ADHD, myelomeningocele, methylphenidate
S ummary
The prevalence of attentional problems, and the e€ ect of
methylphenidate was evaluated in a clinic population of chil-
dren with myelomeningocele. Families of 79 children between
the ages of 6 and 15 years were screened f or the presence of
attention problems in their children, using Conners’ question-
naires for parents and teachers, and/or the DSM-IV checklist.
Thirty-nine per cent of the children exhibited attention prob-
lems, primarily without hyperactivity. Fourteen children with
attentional problems were enrolled in a double-blind placebo-
controlled trial methylphenidate. R esponse to methylpheni-
date was assessed with Conners’ questionnaires, Conners’
Continuous Perf ormance Test, and a battery of selected neuro-
psychological tests. No statistically signi® cant response was
measured for the group while on methylphenidate. Four
children were clinical responders to methylphenidate. The
prevalence of attentional problems in children with myelome-
ningocele is high, and e€ ective medication therapy needs to be
studied further.
Introduction
Professionals who work with children with myelo-
meningocele (MM ) recognize that many of the children
have short attention spans and a high degree of distract-
Authors: Michael Davidovitch, Patricia Manning-Courtney,
Jennifer Watson, Sonya Oppenheimer, Department of
Pediatrics; Linda A. Hartmann, Department of Psychology;
and Marlene Lutkenho€ , Department of Nursing, University
A  liated Cincinnati Center of Developmental Disorders,
Children’ s Hospital Medical Center, Cincinnati, Ohio, USA .
* Author for correspondence. Current address: The Hannah
Khoushy Child Development Center, The Technion, Faculty
of Medicine, Bnai Zion Medical Center, POB 4940, Haifa
31048, Israel. e-mail: davidom@ netvision.net.il
Pediatric Rehabilitation ISSN 1363± 8491 print/ISSN 1464 ± 5270 online Ñ 1999 Taylor & Francis Ltd
http://www.tandf.co.uk/JNLS/pdr.htm
http://www.taylorandfrancis.com/JNLS/pdr.htm
ibility and impassivity [1, 2]. Di culty in sustained
attention to task and focusing can occur in any range
of intelligenc e in children with MM, and some have
these de® cits without any learning disabilities , or have
lesser degrees of physical impairment [3]. Children with
MM showed de® cits across four elements of attention
(encode, sustain, focus/execute and shift) compared with
siblings [4]. Children with MM and hydrocephalu s have
been found to be more distractible than normal controls,
when matched for mental age, during non-verba l tasks
and comprehension of relational words tests [5]. These
authors concluded that the greater distractibilit y is
partly responsible for de® ciencies in vocabular y compre-
hension observed in children with MM. On the Gordon
Diagnostic System (a computerized continuou s per-
formance test) , children with MM of all ranges of
intelligenc e scored signi® cantly above the normal
population , indicating greater inattentio n and impulsiv-
ity [6]. Memory function of children with MM and
shunted hydrocephalus has been evaluated by measuring
the amount of information learned over a number of
trials and the length of retention of this information
[7]. When compared with a control group matched for
Intelligence Quotient (IQ) , children with MM had a sig-
ni® cantly poorer performance on a `Memory for Words’
test.
The extent of disabilitie s associated with MM, as per-
ceived by parents of children with MM, was reported
recently [8]. The parents of 55 children with MM parti-
cipated in the study. With respect to learning, 42 chil-
dren were reported to have learning di culties 26 were
reported to have di culty with concentrating, 21 with
memory, and 16 with organization . Other reports have
described organizationa l problems in children with
MM, and these may involve expression of thoughts,
placement of belongings , or paperwork [9].
 M. D avidovitch et al.
A ttention de® cits may be associated with learning
problems in children with MM. It was postulated that
reducing distractibility , through the use of stimulants in
children with MM may increase learning behaviour s
[10]. However, there are no placebo-cont rolled blind
studies of medication e€ ect on attentiona l problems spe-
ci® c to children with MM. In a placebo-cont rolled study
of several groups of children with neurodevelop mental
disorders, includin g seven children with MM, all were
felt to have responded well to methylpheni date (MPH )
[11]. Signi® cant improvement was reported in 29 of 31
children with MM with attentional problems, during a
non-placeb o controlled, non-blinde d trial of MPH [12].
Much of the published data regarding attention, dis-
Dev Neurorehabil Downloaded from informahealthcare.com by UB Magdeburg on 10/27/14
tractibility and hyperactivit y in children with MM is
based on experience of teachers and parents, and not
on speci® c criteria used to evaluate de® cits in these
areas. This double-blind , placebo controlled study was
designed to estimate the prevalence of attentiona l prob-
lems in a clinic populatio n of children with MM, using
standard measurements, and assess responses to MPH
treatment, using questionnair es and neurophycho logical
testing.
For personal use only.
M ethods
A ll study participant s were recruited from the
Cincinnati Myelomeningocele Program, which consists
of 213 active participants . One hundred and seventy
three (80% ) of these children have shunts. Ninety four
children and adolescents (50 girls) were between the ages
of 6 and 15 years. Parents of 66 children and adolescents
in this age group, who attended the clinic between July
1995 and January 1996, were questioned regarding the
presence of symptoms of inattention , hyperactivit y and
impulsivit y in their children by two of the authors (MD
or PMC ). Parents expressing concerns were given the
Conners’ R ating Scales for parents (CPR S ) to complete,
and were also given the Conners’ R ating Scales for
teachers (CTR S ), to be completed by the teacher.
Children were included in the study if they exhibited
signi® cant elevation (t-score > 65, 1. 5 SD above the
mean ) in one or more of the following categories: impul-
sive± hyperactivit y or hyperactivit y index on the CPR S;
or hyperactvity , inattentive± passive, or hyperactivit y
index on the CTR S. Parents of study participant s were
also questioned using the Diagnostic and Statistical
Manual of Mental Disorders (DSM IV ) [13] checklist
of A ttention De® cit Hyperactivity Disorder (A DHD )
symptoms, to further verify the diagnosi s of attention
de® cits. Children with MM score below the populatio n
average on intelligenc e and achievements tested, but
30
within the normal range. The greatest de® cits occur on
Performance IQ, arithmetic achievement, and visual-
motor integratio n [14]. Therefore, verbal IQ scores
were used and obtained from previous testing informa-
tion, if performed within 3 years of the study. Those
without previous or recent IQ testing underwent testing
with the Wechsler Intelligenc e Scale for Children, 3rd
Edn. Three children with known Verbal IQ of less
than 60 were arbitraril y excluded from the study.
Participants were enrolled in a double-blin d placebo
controlled cross-over design trial of one dosage of
methylphen idate (MPH ) and placebo. Participants
who were already receiving MPH prior to the study
were discontinue d for at least 1 month prior to partici-
pation in the study. Participants were randomized to
receive either MPH or placebo in the ® rst week.
Morning and lunchtime doses were determined by par-
ticipants’ weight, using the following scale: < 20 kg,
5 mg; 20± 29 kg, 7. 5 mg; > 30 kg, 10 mg. A fter seven con-
secutive days of treatment, `medication’ was discontin-
ued for a 1 week `washout’ period. The followin g week,
participant s received the alternate `medication’ , for 1
week. `Medication’ was administered by the parent in
the morning and on the weekends, and by the school
nurse af ter lunch. Evaluation s took place over three
consecutive weeks, and comprised four 1 hour sessions.
The ® rst evaluatio n was performed prior to the start of
`medication’ , as a baseline. The second session took
place af ter the ® rst week of `medication’ , within 1± 2
hours of the participan t taking MPH or placebo. The
third session followed the `washout’ week, as a second
baseline. The fourth session followe d the second week of
`medication’ , again within 1± 2 hours of MPH or pla-
cebo. Evaluation s consisted of the following tests: the
Conners’ Continuou s Performance Test [15], the R apid
A utomatized Naming Test [16], the Purdue Pegboard
(Lafayette Instrument Co., Indiana ), the Digital
Electronic Finger Tapping Test (Western Psychologica l
Services, L.A . ) and the Sentence R epetition Test
(adapted from Spreen and Benton, Victoria ) .
Participants of 10 years and older were also admin-
istered the Symbol Digit Modality Test [17] and Trails
A and B [18]. A ll tests were administered by one of two
authors (MD or PMC ) . Conners’ R ating Scales for par-
ents and teachers, and a Stimulant Drug E€ ects R ating
Scale [19] were completed and collected at baseline, and
af ter each week. The authors were blinded to the results
of the subsequent parent and teacher Conners’ R ating
Scales until all testing was completed.
To further determine the prevalenc e of attentiona l
problems in the clinic population , one author (JW )
attempted phone contact for two groups of families.
 AD HD and m yelomeningocele
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For personal use only.

Figure 1 Flow chart of study participants.

The ® rst group consisted of 41 families, out of the orig-
inal 66 questioned in the clinic during the designated
time period, who did not express concerns regarding
attentiona l problems in their child. The second group
consisted of 25 families who had not been seen in
clinics during the designated time period, and there-
fore not questioned regarding their concerns. Those
contacted were screened regarding attentiona l problems
in their child using the DSM-IV checklist. See ® gure 1
for further description of the study populatio n numbers.
Questionnair e scores and neuropsycho logical test
results were entered into a computer and reviewed for
possible errors and non-norma l distributions . A nalysis
of varianc e for crossover design was used to test the
hypothesis that MPH treatment would result in
improved performance on the various measures.
Sixty-six children (39 girls) in the appropriat e age
group and with verbal IQ above 60 were in clinic during
the study period. Parents of 25 children expressed con-
cerns regarding attentiona l problems in their children,

31
 M. D avidovitch et al.

Table 1 Characteristics of participants in the study

Patient Age (y/m )/Sex L evel of MM Shunt V erbal IQ CPRS* C TRS* DS M-IV CPT+ +

1 7y 1m/M Mid Yes 88 Y es Yes Combined² Inattention

2 7y 7m/F High Yes 86 Y es Yes Inattentive ³ Inattention
3 7y 9m/F Mid Yes 63 Y es Yes Combined Inattention
4 8y/M Low Yes 79 Y es No Inattentive Inattention
5 8y 6m/F High Yes NA No No Inattentive Impulsivity
6 9y 7m/F High Yes 82 Y es Yes Inattentive Inattention
7 10y 2m/F Low Yes 91 Y es Yes Inattentive Inattention
8 10y 4m/M Mid Yes 95 Y es Yes Inattentive Inattention/impulsivity
9 11y 1m/F High Yes 83 Y es Yes Combined Inattention/impulsivity
10 11y 9m/F Low Yes 74 No Yes Inattentive Inattention
11 12y 6m/M Low Yes NA Y es Yes Inattentive Inattention/impulsivity
12 13y 2m\F Low No 127 Y es Yes Inattentive A verage
13 13y 7m/F Mid Yes 76 Y es No Inattentive Inattention
14 14y 4m/M Mid Yes 99 Y es Yes Inattentive Inattention/impulsivity

* CPRSÐ A ttention problems according to Conners’ Ratings Scales for parents.

Dev Neurorehabil Downloaded from informahealthcare.com by UB Magdeburg on 10/27/14

** CTR SÐ Attention problems according to Conners’ R atings Scales for teachers.

² InattentiveÐ Positive DSM-IV criteria for Attention De® cit/Hyperactivity Disorder, predominantly inattentive type.
³ CombinedÐ Positive DSM-IV criteria for A ttention De® cit/Hyperactivity Disorder, combined type.
+ + Conners’ Continues Performance Test Computer ProgramÐ Inattention pro® le; Impulsivity pro® le.
NA Ð Not available.

and were given the CPR S and CTR S questionnaire s. A ttentiveness (d ) category ( p = 0.04) . Despite the lack
0

Nineteen sets of questionnaire s were returned, and 16 of statistically signi® cant improvement for the group,
children met criteria for attentiona l problems. Two of three of the participant s (nos. 7, 9, 14, see table 1 )
the 16 children did not participat e in the study due to
For personal use only.

were judged to be clinical responders to MPH, based

distance from the clinic. Fourteen (nine girls) partici-
pated in the study and their characteristics are detailed
in table 1. Verbal IQ scores were available for 12 parti-
cipants and average verbal IQ score was 88.6. Two par-
ticipants without available IQ scores were estimated to
be functionin g in the average range. Two participant s
had been treated with MPH prior to this study (nos. 7
and 8 ).
Results
A ll 14 participant s completed the trial. For the study
group, analysis of variance for cross over design showed
no statistically signi® cant improvement on any of the
® ve indices of inattentio n or hyperactivit y of the
Conners’ parent and teacher questionnaire s, during
MPH treatment (analysis of the remaining ® ve
Conners’ questionnaire s indices re¯ ecting conduct and
learning also did not show statistically signi® cant
improvement ) . A nalysis of all 13 neuropsycho logical
variables revealed statistically signi® cant improvement
for the group while on MPH only on the colour naming
portion of the R apid A utomatized Naming Test
( p = 0.01) . Of the 12 categories comprising the
Conners’ Continuou s Performance Test, statistically
signi® cant improvement was demonstrated only in the
on improved t-scores of at least 0.5 SD in one or more
categories on both CPR S and CTR S. For these three
participants , additional comments by the parents,
teachers and children also supported the improvements
on the questionnaire s. These same three also demon-
strated improvement on portions of the R apid
A utomatized Naming Test. Participant no. 9 improved
on Trails B, and Participant no. 7 improved on Symbol
Digit Modality Test. A fourth participan t (no. 12 )
improved only on the Conners’ R ating Scales for par-
ents.
Following completion of the study, these four parti-
cipants have been maintaine d on MPH, and a 3 month
follow-up of these participant s indicated continue d per-
ceived bene® t from medication. Two participant s (nos. 6
and 8 ), who did not demonstrate improvement during
the study, were also continue d on MPH by the private
physician , because parents desired a longer trial period,
and they were reported by parents to have bene® ted
from MPH.
Three participant s (nos. 2, 6, 11 ) who received MPH
in the ® rst week of the trial exhibited a response pattern
of initial improvement on MPH, according to Conners’
questionnair es, but continued to demonstrate the same
degree of improvement during the washout and placebo
weeks. No signi® cant side e€ ects were reported during
the MPH or placebo trial, except for excessive crying in

32
 AD HD and m yelomeningocele
one girl (no. 3) . No participant s had to have medication
discontinue d during the trial.
Of the 41 families who came to the clinic but did not
express concerns regarding attention problems, phone
contact was achieved in 27 (the remaining 14 could
not be reached by phone) , all of whom were admin-
istered the DSM-IV checklist. Seven children met cri-
teria according to this checklist for A ttention De® cit/
Hyperactivity Disorder Predominantly Inattentive
Type (® gure 1) . Of the 25 families whose children were
not seen in clinics during the designate d time period,
phone contact was achieved in 13, all of whom were
administered the DSM-IV checklist. Seven children
met criteria for A ttention De® cit/Hyperactivity
Dev Neurorehabil Downloaded from informahealthcare.com by UB Magdeburg on 10/27/14
Disorder Predominantl y Inattentive Type. One child
met criteria for the Combined Type (® gure 1 ).
When the results of the clinic screening and the phone
survey are combined, a total of 79 families were screened
for attentiona l problems in their children (® gure 1 ). Of
these 79, 31 children (39.2% ) were found to be exhibit-
ing attentiona l di culties. Sixteen were found to have
attentiona l problems according to Conners’ R atings
Scales and DSM-IV checklist, and 14 of these partici-
pated in the MPH trial. A n additiona l 15 were found to
For personal use only.
have attentiona l problems according to the DSM-IV
checklist via phone survey. Twelve of the 91 families
of children eligible for this study could not be screened
in either clinic or via phone survey for attentiona l prob-
lems in their children.
Discussion
The prevalence of attentional problems in children
with MM has not previously been reported, althoug h
many clinicians , educators and parents suggest that
there is a higher degree of these di culties (1, 20 ) .
Supportiv e of the above observation , the study revealed
a prevalence of attention problems in almost 40% of
the children , between the ages of 6 and 15, available
for screening. The prevalence of attentiona l problems
in this populatio n is much higher than the 3± 5% that
is reported in the general population (A merican
Psychiatric A ssociation 1994 [13]. Furthermore, this
prevalence may represent an underestima te of the true
prevalence of attentiona l problems in this population , as
teachers were not screened in this process. A ttentional
problems in children with MM may provide an addi-
tional obstacle to learning, as well as a€ ect the child’ s
ability to remember to perform tasks of daily living,
such as catheterization. Because of the multiple medical
complication s some of these children experience,
families of children with MM may not prioritize their
child’ s attentiona l di culties, or simply not recognize
them. Some parents are also reluctant to treat their
children with an additiona l medication, and this may
cause some to be hesitant in acknowledging problems
that may require additiona l medical therapy. A s demon-
strated during the phone survey, seven children whose
families denied attentiona l problems when questioned
during the clinic visit met criteria for A ttention
De® cit/Hyperactivity Disorder Predominantly
Inattentive Type on the follow-up phone survey.
Seventy ® ve per cent or more of children with
A ttention De® cit Hyperactivity Disorder, without
MM, bene® t from MPH treatment [21, 22]. The
response rate of children with MM and attentiona l def-
icits in the study was much lower, with only four of 14
participant s judged to be clinical responders. There are
several possible explanation s for this lower response
rate. First, the small number of study participant s may
have hindered the ability to achieve a statistically signif-
icant response rate. Secondly, while this dosing regime,
which provided an expected per kilogram dose of
0.3 mg, has been demonstrated to be most bene® cial in
improving attention and memory function [23, 24] it is
possible that a higher dose in some of the participant s
would have been bene® cial. In one report [11], in which
seven children with MM responded to MPH, MPH
dosage was titrated up in participant s until a desired
e€ ect, or adverse e€ ect, was observed. A third possibility
may be that the underlyin g aetiology of attentiona l
problems in children with MM is di€ erent from the gen-
eral population , and re¯ ective of the structural brain
abnormalitie s in children with MM, which may account
for a di€ erent response rate to MPH.
The participant s in this study who underwent the
MPH trial were assessed with the Continuou s
Performance test (CPT) , a battery of neuropsycho logical
testing, and questionnaire s. In this study, only question-
naires were found to be useful indicators in assessing
individua l response to MPH. Thirteen of the MPH
trial participant s exhibited attentiona l di culties and/
or impulsivity , according to the CPT (table 1) , which
further supports the diagnosis of attention di culties
and/or impulsivit y according to the CPT (table 1 ) in
these participants . However, the CPT was not helpful
in assessing response to MPH. Signi® cant improvement
was demonstrated on only one category of the CPT for
the group during MPH treatment. This category,
A ttentiveness (d ) , is not one of the categories that is
0
speci® cally re¯ ective of attention or impulsivit y [15].
Children with MM are known to have di culties with
® ne motor and visual± motor coordinatio n [25] and it is
posible that attentiona l problems may compound these
33
 M. D avidovitch et al.
di culties. For this reason, the neuropsychol ogical tests
used in this study were performed to measure possible
changes in these areas, during MPH trial. Signi® cant
improvement was demonstrated only on the colour
naming portion of the R apid A utomatized Naming
Test. While this change represents improvement in
assignin g a verbal response to a visual stimulus, the
other neuropsycho logical testing provided no additional
evidence that MPH improves ® ne motor and/or visual
motor coordination in this group of children.
Children screened tended to exhibit more inattention
and less hyperactivit y according to the DSM-IV check-
list, as was reported previously [26]. It is not clear
whether these children are actually not hyperactive, or
Dev Neurorehabil Downloaded from informahealthcare.com by UB Magdeburg on 10/27/14
simply not perceived as hyperactive, possibly because of
their physical disabilities . Measures of hyperactivity are
di cult to utilize for the child in a wheelchair. Children
with MM may exhibit a high frequency of super® cial
talking, which was also observed in the study popula -
tion, and this may replace more common symptoms of
hyperactivity [20]. Lack of hyperactivit y in this popula -
tion may make it less likely for the teacher to report
attentiona l problems in these children.
In conclusion , the prevalence of attentiona l problems
For personal use only.
in children with MM is high, and these of ten occur in
the absence of hyperactivity . Therefore, it is important
to routinely screen children with MM for attentiona l
problems. If these exist, medication management can
be considered. Further studies should address higher
doses of MPH. Trials should be conducted in a double
blind placebo controller manner, to assure e€ ectiveness
of medication therapy. Children with MM are also can-
didates for non-medical interventions , such as counsel-
ling, classroom modi® cations, for attention problems,
and these should be discussed with the teacher and
family.
Acknowledgements
This project was supported by the Spina Bi® da
A ssociation of Cincinnati , and by the Project No.
MCJ-339156-06-0, awarded by the Maternal & Child
Health Bureau and Health R esources and Services
A dministratio n, Public Health Service, Department of
Health and Human Services (DHHS ) and A ward No.
90DD0328/03, awarded by the A dministratio n on
Developmental Disabilities , A dministration for Child
and Families, DHHS.
We wish to thank the children and families participat -
ing in this study and Dr J. H. R ubinstein for his help in
the preparatio n of this manuscript.
34
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