A
TWENTY-FIVE
YEAR
FOLLOW-UP
STUDY
ON
THE
HYPERKINETIC
CHILD
WITH
MINIMAL
BRAIN
DYSFUNCTION
Miriam M. Menkes, M.D., Jane S. Rowe, M.D., and John H. Menkes, M.D.
From the Departments of Pediatrics ansi Division of Neurological Medicine, Johns Hipkins Hospital
and the Maryland Institute for Children, Baltimore, Maryland
(Received June 23; revision accepted for publication September 19, 1966.)
The study was supported in part by grants from the United Cerebral Palsy Research and Educational Foundation and grants NB 05212 and 5 Ti NB-5359-04 of the U.S. Public Health Service.
J.H.M. was Joseph P. Kennedy, Jr., Memorial Foundation Scholar in Mental Retardation.
PRESENT ADDRESS: (J.H.M.), Division of Pediatric Neurology, University of California at Los Angeles,
Los Angeles, California.
PEDIATRICS, Vol. 39, No. 3, March 1967
393
MONG the large number of children
pre-senting with behavior and learning
dis-orders, there is a percentage with clinical
manifestations sufficiently distinct so that
they may be set apart as showing a definite
syndrome commonly termed “the brain
damaged” child. The typical patient is
hy-peractive and easily distractible, has a short
attention span, and is emotionally labile,
easily frustrated, and impulsive. Due to
these factors and also due to a specific
learning disorder, leading most commonly
to difficulty with abstraction, classification,
spatial relationships and pattern
percep-tion, his school work is poor-more so than
would be expected from his I.Q. score.
Such children were first described in 1941
by Werner and Strauss’ who felt that the
syndrome revealed brain injury and who
distinguished it from “simple” mental
retar-dation. More recently, this group of
pa-tients has been variously termed “minimally
brain damaged,”2 “minimal chronic brain
syndrome,”3 “hyperkinetic behavior
syn-drome,” and “minimal brain
dysfunc-tion.” While most of the patients in the
initial report had fairly convincing
histori-cal and clinical evidence for perinatal or
infantile cerebral injuries, and all were
re-tarded, more recent descriptions of such
children have tended to exclude those with
neurologic defects so gross as to
over-shadow other problems they might have.
This has led to a considerable amount of
confusion since different authors have used
different criteria as evidence for the
pres-ence of brain dysfunction. Some, as for
in-stance Strauss6 and Laufer and Denhoff,4
feel that the behavioral syndrome is
charac-teristic enough to be diagnostic by itself,
while others emphasize that it may be
du-plicated in emotional disturbance, and that
the diagnosis must be supported by
evi-dence for brain dysfunction in the history
or 78 These problems
of terminology and differential diagnosis
have been reexamined by Work and
Haldane in the light of recent
neuro-physi-ologic developments.
Paine,3 by careful clinical examination of
a group of children with a typical pattern
of behavior and learning problems, found
definite neurological abnormalities in
al-most all. These included mild
choreoatheto-sis, hyperrefiexia, tremor of hands, and
varying degrees of dysarthria.
Our experience has been in agreement
with this, and we feel that a mild, chronic
brain syndrome in children with minimal
detectable affection of motor, mental, and
sensory functions represents a valid clinical
entity. At the present time, little
informa-tion as to the natural history of this disorder
is available. This is partly because only
within the last 25 years has this syndrome
become separated from other learning
dis-orders. We have, therefore, undertaken a
long-term, follow-up study of children
whom we have diagnosed retrospectively as
child with minimal brain dysfunction.
Specifically, we have aimed to determine
what level of social-functioning is achieved
by these children and what factors can be
selected as being of value in predicting the
functional outcome.
ORIGINAL PATIENT MATERIAL
Eighteen cases fitting the following
diag-nostic criteria were selected from the
rec-ords of The Johns Hopkins Hospital Child
Psychiatry Out-Patient Clinic where they
had been seen on one or more occasions
during the years 1937 to 1946.
All had presented with hyperactivity and
learning difficulties and showed evidence of
one or more of the following
non-behav-ioral criteria for brain dysfunction :
clumsi-ness of fine movements, visual-motor
deficits, impaired or delayed speech.
Under “hyperactivity” we included the
accompanying elements of distractibility,
short attention span, emotional lability,
im-pulsivity, and low frustration threshold.
Patients with seizures or with I.Q. levels
of less than 70 were excluded. None of the
children had been diagnosed as being
psy-chotic.
Detailed records were available on all
patients. Usually these contained a wealth
of data concerning the child’s personal,
medical, social, and family history as well
as a psychiatric examination, Binet test
form L or M, and either a physical
exam-ination or many pertinent descriptive
de-tails. In all but one instance, no
electroen-cephalographic tracing was performed.
From information in the chart, patients
were classified as showing either “definite”
or “probable” evidence of brain
dysfunc-tion. Evidence was considered to indicate
“definite” brain dysfunction if more than
one of the non-behavioral criteria was
pres-ent as well as the behavioral symptoms.
Ten patients fell into this group. Those
hay-ing only one of the non-behavioral criteria
were classified as “probable brain
dysfunc-tion.” There were eight of these.
Table I summarizes information about
the original sample of 18 patients. Of these,
14 were male and four were female. All
were white. The age at the first clinic visit
ranged from 2 7/12 to 15 6/12 years with a
mean of 7 0/12 years. Fourteen patients
were first seen between 5 and 10 years. We
assigned a social class rating* to each
pa-tient based on the occupation of the
pa-tient’s father at the time of the out-patient
visit.’0
As shown in Table I the parents of four
children were in social Class 1, one was in
Class 2, four were in Class 3, two were in
Class 4, one was in Class 5, and six were in
Class 6. Six patients originally scored I.Q.
over 100, five scored between 90 and 99,
three scored between 80 and 89, and four
scored between 70 and 79. We attempted
also, on the basis of a wealth of descriptive
material in the charts, to assess the patient’s
home environment with particular attention
to the family as a functioning unit and the
attitude of the child’s parents toward him.
On this basis the patients fell into two very
distinct groups, with no overlapping. The
home environment was classified as
“favor-able” when the families were stable, and
family members appeared affectionate
to-wards each other and the patient. Five
fam-iies answered this description. The
remain-ing 13 were termed unfavorable. In all of
these, relationships of the family members
to each other had been described in
strong-ly derogatory terms ranging from
“marked-ly aloof and cold” to “open hatred.” In eight
of these, the family had already
dis-integrated or did so shortly after first
com-ing to the clinic; four of these were
de-scribed as “constantly chaotic.” Ten of the
patients had been referred to the clinic by
the school, one by a private physician, and
seven others were self-referrals.
Manage-ment varied widely within the group. Four
were seen only once, for diagnostic
evalua-tion by the Director of the clinic; three
pa-0 The following ratings, based in part on
Dril-lien’s classification, were used: Class
1-profes-sional; Class 2-intermediate; Class 3-skilled
occu-pation; Class 4-partly skilled occupation; Class 5
-unskilled laborer; Class 6-unemployed father,
ARTICLES
TABLE I
SUMMARY OF CLINICAL DATA ON 18 PATIENTS* WITH HYPERKINESIS AND MINIMAL BRAIN DYSFUNCTION
SELECTED FOR Fou.ow-up STUDY
.
Patient Sex
Age
first
seen
Brain Dy
Definite
.1efnnction
Probable
#{149} Binet
I.Q.
Evoiua-.
lion
ofliome
Soda-.
economic
Class
Referral
Source
Follow-up
J.B. M 8h + 128 F 1 family E
P.E. M 6 + 115 U 1 school E
M.P. M 15 + 104 U 4 school E
F.C. M 6 + O3 U 5 school I
w.C.
1-1 7?, + 1o U 3 school EilK. M
8-
+ 101 U 6 school EAll. M 9i + 95 U 1 school I
LB. F , + 93 U 6 family I
CR. M 8 + 91 U S school E
C.M. M 4, + 88 U 1 family E
HG. M 7, + 84 F S family E
C.R. F 6 + 76 F 4 family E
O.M. F
W1
+ 72 U 6 school ES.L. M sfl + 71 U 6 physician E
T.T. M 8 + 96 U 6 school 0
AL. M 11ff + 95 F 3 school 0
M.D. M 7k-f + 84 U 6 family 0
ML. F 935 + 73 F 2 family 0
* All the patients were white.
F-favorable; U-unfavorable.
E-neurologic and psychologic examination; I-interview only; 0-no follow-up information.
tients were seen on two occasions. The rest
were followed fairly consistently for
peri-ods varying from 2 to 7 years. None of the
patients received drug therapy. Those who
returned to the clinic received support and
encouragement for parent and child,
to-gether with efforts aimed at manipulating
the environment (particularly school and
the wider family) toward better meeting
the child’s needs.
FOLLOW-UP STUDIES
Of the 18 patients whose charts were
mi-tially selected, 14 were available forfollow-up; 11 of these were examined by at least
one of us. Examination included an
inter-view to elicit the patient’s personal and
medical history in the intervening years as
well as his present mental and
socioeco-nomic status, neurological examination, and
administration of a recognition vocabulary
test (Ammons Full Range Picture
Vo-cabulary)h1 and the Bender Gestalt test.
Of the three remaining patients, one was
interviewed by telephone and information
about the other two was gained in
inter-views with their mothers.
The follow-up information is summarized
in Table II.
The time that had elapsed since the
pa-tients were originally seen in the clinic
ranged from 14 to 27 years, with a mean
follow-up period of 24 years. At the time of
reexamination, four patients were in
insti-tutions, diagnosed as psychotic; two were
clearly retarded and living dependent lives
with their families; eight were self
support-ing. Of these eight, four have in the past
spent some time in an institution : two in
institutions for delinquent boys, one in a
hospital for the retarded, and one in jail.
Of the 11 patients examined
neurologi-cally, eight had definite evidence of
neuro-logical dysfunction, one had equivocal
evi-dence, and two had none. In most cases,
the abnormalities found included terminal
intention tremor and minimal
alterna-396
TABLE II
SUMMARY OF FoIIow-uP INFORMATION OBTAINED ON 14 PATIENTS WITH HYPEHKINESIS AND MINIMAL BRAIN
DYSFUNCTION : PRESENT SOCIAL ADJUSTMENT
. Patient Age at Fol- low-up Fol- low-fl/) I nter-cal (yr) Age Restlessness . Disappeared . Neurologic Abnor-. . inahties F ocab-neary ‘ est Score Bender Perform-ance . In.sti-. tution-. ahzed , . P amily Sup-ported Self- Sup-ported . Past History . of Institti-. . tionalization J.B. P.E. M.P. F.C. w.C. H.K. All. LB. CR. C.M. B.C.
cit.
O.M. S.L. ‘35 31 40 31 30 33 8 8 22 Si si 29 33 30 7 5 25 5 3 25 19 5 14 26 24 3 4 24 14-15 14 12 110 14 yes, when? yes, when? IiO 14 8 1 no yes, when? definite probable none not seen definite definite not seen not seen definite definite definite definite definite none 122 105 68 -106 86 -76 110 60 57 78 103 below avg. below avg. poor -below avg. poor -below avg. poor poor very poor poor good x x x x x x x x x x x x x x MTS MTS, CSC jail ltSlTMTS-Maryland Training School for Boys; CSC-Child Study Center (a residential facility for PsFclliatric
treatment of children); RSH-Rosewood State Hospital (institution for the retarded).
ting movements and impaired tandem gait.
One patient (J.B.) had more extensive
neu-rological abnormalities, including a partial
expressive and receptive aphasia and a
rest-ing tremor. In addition to these findings,
four patients had moderate and six patients
severe visual motor impairments as
re-vealed by the Bender Gestalt test.
Three of the patients complained that
they still felt restless, had a hard time
set-tling down to anything, including a
televi-sion show, and changed jobs frequently. In
the other 11, signs of hyperactivity had
dis-appeared; three did not remember when
this was. In the eight remaining patients,
this had happened at ages ranging from 8
to 21 years; in six of them it was between
12 and 14 years.
We were unable to find any correlation
between the patients’ present social
adjust-ment and the classification of their early
home environment as favorable or
unfavor-able. This is not surprising since the
con-cepts “favorable” or “unfavorable”
environ-ment-as well as “psychotic,” “dependent,”
“self supporting”-are all global, each
representing the sum total of a very large
number of variables; our sample, on the
other hand, is small. We suspect that in
order to elicit prognostic factors in the
home environment a much larger sample
would have been needed. Also, individual
aspects, both of environment and the
pa-tient’s eventual functioning, would have to
be more specifically identified and
de-scribed. Similarly, we found no correlation
between the outcome and the amount of
treatment the patient had received. Here,
once again, our sample is small and the
number of variables large; it was apparent
on reading the charts, for instance, that
sev-eral of the families attending the clinic over
a long period of time did so because they
had formed relationships with personnel
there which they felt were helpful. Others,
on the other hand, were returned again and
again over the years by exasperated social
agencies such as schools, public welfare, or
the court. Our sample was too small to try
TABLE III
CORRELATION OF I.Q. SCORE ON INITIAL
EvAIuA-TION WITH LATER SOCIAL ADJUSTMENT
\Vhile it is not possible to make a strict
comparison between the scores achieved
during childhood on a Stanford-Binet test
and a subsequent Ammons test, it is
inter-esting to note that, although all patients
Se-lected for follow-up studies had initial
I.Q.’s over 70, the follow-up I.Q. was within
10 points of the original score in only four
of the 11 patients retested. Three of these
four were those with the highest scores. In
five patients, the follow-tip I.Q. was more
than 10 points lower than the initial I.Q.; in
two it was 10 points higher (these two are
in the group who are at present psychotic).
The other two in this group are among
those scoring more than 10 points lower
than originally. In the case of B.G. and
G.R. it is likely that a good environment
originally led them to gain a higher
Stan-ford-Binet I.Q. than justified by their later
achievements or test results. When the
orig-inal I.Q. score is correlated with the
assess-ment of brain dysfunction, it is apparent
that all but one of the patients whose
origi-nal I.Q. scores were over 100 fall into the
probable brain dysfunction group, and all
but one of those with scores below 100 fall
into the definite brain dysfunction group
(Table I). This helps to provide validation
of the diagnosis, since the I.Q. test
perfor-mance was not in itself part of the
diag-nostic criteria. Also, this finding tends to support the assumption that “minimal
dys-function” is associated with lowering of the
I.Q. even though it may remain within the
“normal” range.
When comparing the subjects’ original
I.Q. with their present socioeconomic status
(Table III), it is seen that most of the
pa-tients with the highest I.Q. scores are at
present self-supporting. Of the nine
pa-tients initially scoring above 90, seven are
self-supporting.
The predictive value of the initial I.Q.
test must be accepted with caution,
how-ever. That it is not the sole factor in
deter-mining ultimate social independence is
shown by O.M., who, despite initial and
follow-up I.Q.’s of 72 and 78, respectively,
is financially independent and maintains
I.Q. Se1f-Supported
Score Number Yes No
Over 10(1
90-99 80-89
70-79
6
3
3
4
3 0 1
0
herself restlessly but with persistence by
moving from job to job around the country.
The Binet test form administered to these
patients is largely a test of verbal skills. We
do not know whether it is the intactness of
the functions that these represent which
make for adequate social adaptation later
in life or whether different abilities are
in-volved here which are not directly
mea-sured by the I.Q. test but vary in parallel
with those that are. Also, since I.Q. scores
correlate broadly with the degree of
“mini-mal dysfunction,” the more severe the
dys-function the poorer the prognosis in terms
of socioeconomic status.
DISCUSSION
In retrospective studies such as ours, it is
important to consider two factors. The
di-agnosis of “minimal brain dysfunction” was
made from information gathered by others
at a time when the syndrome had not been
delineated. Thus, electroencephalographic
tracings, which nowadays are usually
in-eluded in the investigation of a suspected
case, were generally not available to us.
Also, regarded as a group, the patients
se-lected for follow-up differ somewhat from
children with minimal brain dysfunction
seen these days in an out-patient clinic.
Al-though the age and male-female ratio was
that generally seen among children with
minimal brain dysfunction, non-white
chil-dren are absent from the original sample.
This reflects the makeup of the clinic
popu-lation at that time. However, diagnosis was
definitely justified in 9 of the 11 patients
the eight patients in the original sample
classed by us as “definite brain
dysfunc-tion,” all six who were reexamined showed
neurological abnormalities. Of the six
pa-tients originally classed as “probable brain
dysfunction,” three of the five reexamined
were found to have neurologic
abnormali-ties. The other two, however, are now
clearly psychotic and have no neurologic
abnormalities. The fact that a number of
children diagnosed by us as having
“mini-mal brain dysfunction” developed
psy-choses may mean that, in part, the diagnostic
criteria used by us were not rigorous
enough to exclude children who presented
a picture of brain dysfunction on a purely
psychiatric basis. On the other hand, it may
well reflect the natural history of the
syn-drome of hyperactivity with minimal brain
dysfunction of childhood. The latter
possi-bility is exemplified by the fact that two
other patients, at present psychotic, were
found on reexamination to have definite
neurologic abnormalities.
Birchll has pointed out that among the
behavior syndromes which may accompany
central nervous system dysfunction are
some which are indistinguishable from the
childhood psychoses. It is likely that
organ-ic disturbances in body boundaries and in
interpretation of sensory input data can
re-sult in a self re-enforcing process of
mala-daptation and alienation leading to a
cmi-cal picture identical with the so-called
“functional” psychoses. Therefore, we
con-elude that the syndrome of minimal brain
dysfunction as it is presently recognized
may stem from one or more different
causes, each of which affects the character
and severity of the clinical picture, the
re-sponse, if any, to various forms of
treat-ment, and the ultimate functional outcome.
SUMMARY
Eighteen patients who had been
evaluat-ed at a child psychiatry clinic a mean of 24
years ago were selected for follow-up by
the following criteria: the presenting
symp-toms had been hyperactivity and short
at-tention span, none had seizures, all had an
I.Q. over 70, and all had indications of
neu-roogic abnormalities such as poor
coordi-nation, visual motor dysfunction, or speech
impairment. Eleven of the 18 were
reexam-med fully and an interview or information
was obtained about three more. Neurologic
examination showed definite abnormalities
in eight subjects and suspicious findings in
one, but it was normal in two.
Hyperactiv-ity was still present in three subjects aged
22 to 23 years and had disappeared
be-tween the ages of 8 and 21 in the others.
Four subjects were institutionalized as
psy-chotic, two others were retarded and
sup-ported by their families, and eight were
self-supporting. Of the latter, four have
spent some time in institutions. A major
prognositc factor was the 1.9. obtained
during initial evaluation; all but one of the
subjects who were self-supporting had
scored an I.Q. above 90.
REFERENCES
1. Werner, H., and Strauss, A. A.: Pathology of
figure-background relation in the child. J.
Abnorm. Soc. Psychol., 36:236, 1941.
2. Knobloch, H., and Pasamanick, B. : Syndrome
of minimal cerebral damage in infancy. J.A.M.A., 170:1384, 1959.
3. Paine, R. S.: Minimal chronic brain syndromes
in children. Dev. Med. Child. Neurol., 4:21,
1962.
4. Laufer, M. W., and Denhoff, E.: Hyperkinetic
behaviour syndrome in children. J. Pediat.,
50:463, 1957.
5. Clements, S. D.: Minimal brain dysfunction in
children. NINDB monograph No. 3. Wash-ington, D.C.: U.S. Department of Health,
Education, and Welfare, 1966.
6. Strauss, A. A., and Lehtinen, L. E.:
Psycho-pathology and Education of the
Brain-Injured Child. New York: Grune and
Strat-ton, 1947.
7. Rapin, I.: Brain damage in children. In
Bren-neman-Kelley: Practice of Pediatrics, Vol. 4.
Chapter 17, Hagerstown, Maryland: W. F.
Prior Co., 1964.
8. Cameron, K., Lewis, M., and Stone, F. H. : Is
there a syndrome of brain damage in
chil-dren? Cer. Palsy Bull., 3:74, 1961.
9. Work, H. H., and Haldane, J. E.: Cerebral
dysfunction in children. Amer. J. Dis. Child.,
111:573, 1966.
fac-tors affecting the incidence of premature
birth. I. Premature births without
complica-tions of pregnancy. J. Obstet. Gynaec. Brit.
Emp., 64:161, 1957.
1 1. Blatt, S. J.: Recall and Recognition Vocabulary.
Arch. Gen. Psychiat., 1:473, 1959.
12. Birch, H. W.: The problem of “brain damage”
in children. In Brain Damage in Children.
Baltimore: Williams and Wilkins Co., 1964.
Acknowledgment
The authors wish to thank Dr. Leon Eisenberg
and Miss Barbara Ashenden for allowing them
ready accessibility of the patient records.
MOTHEIIS OF RETARDED CHIumN: How
THEY FEEL: WHERE THEY FIND HELP, by
Walter H. Ehlers. Springfield, Illinois:
Charles C Thomas, 1966, 161 pp., $6.75.
This book comes when there is great
activ-ity in the field of mental retardation and when
a study such as this is immediately pertinent.
As the sample for his study, Dr. Ehlers selected
24 mothers from the files of the Cambridge
Service for Retarded Children, a public health
clinic in Cambridge, Massachusetts. The
au-thor not only explored the mothers’ feelings and attitudes about their child’s retardation
and how and why they sought help, but he cx-amined the functions of the clinic and its eight
workers. How the interrelationships between
team members and the overlapping function and unresolved differences of opinion regard-ing problem management could and did
inter-fere with the effectiveness of the clinic’s service
is emphasized throughout the book.
In more than a decade of work with parents
of retarded children seen at the Children’s
Hos-pital Medical Center in Boston, the reviewer
has met most of the situations described in this
book. In some aspects, our experiences differ.
This may be due to the kind of sample used by
Dr. Ehiers and to the difference between the
methods of operation of the Cambridge
Ser-vice for Retarded Children and that of the
Boston Children’s Hospital. The reviewer
worked closely with the Cambridge agency
since its inception; she-rather than the
physi-cian-was responsible for all the referrals to
the agency (9 out of 24 mothers in the study).
This point is made because the majority of
physicians apparently have only sketchy ideas
of community resources for the retarded child.
This book is written with warmth and
com-passion; those of us who have worked in the
field of mental retardation will note with a
pang how many times we were less than
sue-cessful in our commitment. Physicians will find
the book helpful in sorting out their own
thoughts and feelings about how they can best
help mothers of retarded children.
