0145-6008/92/1603-0558$3.00/0 ALCOHOLISM:CLINICAL A N D EXPERIMENTAL RESEARCH
Vol. 16, No. 3 May/June 1992
Autism in Fetal Alcohol Syndrome: A Report of Six Cases J. L. Nanson
This paper reports on six children who all fulfill the criteria for fetal alcohol syndrome and have a history of maternal alcohol abuse during pregnancy, and who also fulfill the criteria for diagnosis of autism. Their behavior and natural history is compared with eight contrast FAS children, of similar ages and functional levels, who were free of autistic behaviors. The autistic FAS children were all moderately or severely retarded. Autistic behavior has not been commonly associated with prenatal alcohol exposure. Clinicians need to be aware of this potentialdual diagnosis so that appropriate educational and treatment resources can be employed. Key Words: Fetal Alcohol Syndrome, Physical Growth, Autism, Mental Retardation.
RENATAL EXPOSURE to excessive maternal alcoP hol ingestion results in a wide variety of defects ranging from life threatening anomalies to subtle problems with learning and attention. Although the variability in physical manifestations of alcohol-related birth defects is well recognized, variability in the behavioral manifestations of prenatal alcohol exposure is less well described in the literature. The purpose of this paper is to describe six children, ranging in age from 6 to 15 years, who fulfill the criteria for a diagnosis of both fetal alcohol syndrome (FAS) and autism. The behavior of these children is very different from previously published descriptions of behavior in children with FAS, particularly in regard to their social and emotional f~ n c t i o n i n g . ~ In-~order to assess the extent to which these children are different from other FAS children, their behavior and social histories are compared with a contrast group of FAS children, matched for age and degree of retardation who did not show the characteristics of autism. The diagnostic criteria for FAS have been revised by the Fetal Alcohol Study Group of the Research Society on Alcoholism (RSA)’ to include prenatal and/or postnatal growth retardation of weight and height, craniofacial anomalies, and evidence of central nervous system dysfunction such as developmental delay, mental retardation, hyperactivity, or tremor. In the literature, reported cases of FAS have varied greatly in terms of the degree of cognitive impairment present, from IQs within the normal range to IQs in the severely retarded range, with mean IQs typically in the mildly retarded or borderline ranges.8 Streissguth and Ladue’ have suggested that there is a ‘7’
From the Alvin Buckwold Centre, Royal University Hospital, Saskatoon, SK, Canada. Received for publication June 20, 1990; accepted December 3, I991 Reprint requests: Dr. JL Nanson, Alvin Buckwold Centre, Royal University Hospital, Saskatoon, SK Canada S7N 0 WO. Copyright 0 1992 by The Research Society on Alcoholism. 558
behavioral phenotype of FAS, with unique manifestations in different developmental levels. In young children, impulsivity, hyperactivity, over-friendliness, poor social judgement, and a sprightly, butterfly-like manner or movement were noted. In school age children, hyperactivity continued to be problematic and to account for some of their significant learning difficulty in school. The authors describe the highly social orientation of these children, that often gives the impression that they are more intelligent than intelligence test scores suggest. Problems with over-talkativeness, over-intrusive behavior, and poor social judgement persist into adolescence and adulthood, and contribute to significant loneliness and few friendships. The children described in this paper manifested the physical phenotype of FAS, and had a history of maternal alcohol abuse during pregnancy but had a very different behavioral phenotype, typical of autistic children, and characterized by more significant retardation of both their cognitive and social skills. In contrast to FAS, which is diagnosed on the basis of growth retardation, dysmorphology and central nervous system (CNS) dysfunction, and is associated with a uniform etiology, autism is diagnosed on the basis of the child’s behavior and the natural history of the disorder, without reference to growth, dysmorphology, or etiology. Although the incidence of dysmorphic features is increased in the autistic population relative to normal individuals, no isolated defect or pattern of dysmorphic features has been reliably associated with autism.’ Similarly, recent work” has failed to isolate a single factor or combination of factors that contributed to the etiology of autism. Although a psychogenic etiology of autism was initially suggested when Kanner” first described the disorder in 1943, subsequent research has failed to support this idea. Current research in the area is primarily focused on neurobiological aspects of the disorder now.” A neurobiological basis for autism is suggested by the increased incidence within some families” and the increased frequency of mental retardation and other symptoms of central nervous system impairment in individuals with autism. Kanner’ I originally thought that autistic individuals were of normal intelligence but often had low scores on intelligence tests because of their defects in social relationships. Subsequent research has failed to support this view, and it is now thought that the majority of autistic individuals are also mentally retarded. For example, Ritvo and FreemanI3 have suggested that 60% of autistic children have IQs below 50, 20% have IQs between 50 and 70, and only Alcohol Clin Exp Res. Vol 16, No 3 , 1992: pp 558-565
AUTISM IN FETAL ALCOHOL SYNDROME
20% have IQs above 70. Thus, mental retardation and autism are separate disorders but frequently co-exist in the same individual. Autistic individuals are also more likely to have uneven intellectual profiles whereas nonautistic, retarded individuals are more likely to have uniform defects in all areas as~essed.’~ Coleman” has suggested that autism is a syndrome with many different etiologies. Chromosomal disorders, such as fragile X; prenatal infections, especially rubella; untreated metabolic conditions, such as phenylketonuria; and other genetic syndromes, such as tuberous sclerosis are also associated with an increased incidence of autism. In almost all of these conditions, the affected individuals are also mentally retarded. The hallmark of autism is a defect in social behavior and attachment. Other defects include problems with both verbal and nonverbal communication, and perseverative, stereotypic and ritualistic behaviors. In spite of general agreement on the core features of the disorder, the diagnostic criteria for the disorder are still a matter of some dispute amongst clinicians. In the Revised Diagnostic and Statistic Manual of Mental Disorders (DSM-I 1 1R),I6autism is classified as one of several disorders originating in infancy and early childhood subsumed under the title of Pervasive Developmental Disorders (PDD). The diagnostic criteria for PDD include both behavioral and historical components. Allen” has argued that all of the pervasive developmental disorders listed in the DSM-I 11R are variants of autism and should be classified as such. She has suggested that the variability in the clinical presentation of autism is a consequence of qualitative differences in social, language, play, and communication skills. SchreibmanI8 has reviewed the behavioral features of autism and listed eight key deficits that clinicians typically use in making the diagnosis of autism. Of these, six are much more common in autism than other disorders, while the remaining four overlap with mental retardation. The six unique features are: I. A defect in social behavior. Autistic children, regardless of their intellectual abilities, appear detached from their social environment. They prefer to be alone, and may ignore or react negatively to affection and social interactions offered by parents and other children. 2. Defects in speech and language. Approximately half of all autistic individuals never develop any meaningful speech,”-” and those that do typically have language use that is qualitatively different from other children. Echolalia (repetition of words and phrases spoken by others), and pronominal reversal (using you for I and proper names for me), as well as defects in comprehension and speech rhythms (prosody) are also character is ti^.^^-*^ 3. The preservation of sameness. This was first identified by Kanner” as a key feature of autism. Autistic individuals are typically very sensitive to minor changes in their environment, such as a change in the location of a toy or
559
a new activity added to daily routines. Often associated with this need for sameness is a ritualistic preoccupation with a single activity such as memorizing maps or train schedules. Ritualistic eating patterns, such as consuming only one food, are also common. 4. Abnormal responses to their sensory environment. Both under- and over-responsiveness to sensory stimulation are reported.25Often these individuals are suspected of being deaf because of their lack of responsiveness to sounds but on examination, have normal hearing. The same individual may show extreme reactions to certain classes of sounds. Similar, variable, and inconsistent responses to taste, touch, and pain are also common.*’ 5 . Exceptional abilities in one isolated area such as music or mathematics.26This skill may represent one area of average functioning in an individual with depressed functioning in a number of other areas or may represent a truly exceptional ability. 6. Emotional responsiveness or affect. This final area is usually identified as abnormal in autistic individuals, regardless of their intellectual abilities. Autistic individuals may show flattening of affect, or have rapid and unpredictable mood swings that appear unrelated to social events. The remaining diagnostic features: self-stimulatory behaviors, such as rocking, hand flapping, and spinning and self-injurious behavior, such as head banging or biting of hands are characteristic of autistic individuals,” but are also common in the mentally retarded, especially those with sensory impairments, such as the deaf-blindI4and in girls with Rett syndrome.25Self-stimulatory behaviors may preoccupy an autistic individual to the preclusion of all other activities. These behaviors are often resistant to treatment. The diagnosis of autism is typically made between the age of 2 to 4 years of age when the affected child fails to develop appropriate speech and social skills. Both the natural history of the disorder and the child’s behavior are used in making the diagnosis. Although a number of abnormalities have been reported as more frequent in autism, there is no single investigation or finding that will confirm a diagnosis of autism. Rather it is the presence of the constellation of symptoms that leads to a diagnosis. Most clinicians do not view autism as a well-defined syndrome with a single etiology but rather as a spectrum of behaviors, which can characterize individuals with very different levels of intelligence. In order to assist with the differentiation of this spectrum of autistic behaviors, a number of different types of rating scales have been developed. One of the more recent of these is the Childhood Autism Rating Scale.27The CARS is a 15-item scale that is designed to differentiate between autistic and non-autistic children, with and without concomitant mental retardation. It is designed to be administered by a psychologist, using the parent or someone else familiar with the child as an informant. The items
560
NANSON
are based on five diagnostic systems that have been proposed for the diagnosis of autism, including the DSM I11 criteria. The items are similar to those described by Schreibman (see above). The CARS is based on studies involving over 1500 children of all ages, both retarded and non-retarded who have been assessed by Schopler and his colleague^^^ in their program for autistic children at the University of North Carolina. Internal consistency, interrater and test reliability are all reported as with acceptable limits for a behavioral rating scale.The validity of this scale was assessed by correlating CARS scores with independent clinical diagnoses of autism. Different samples yielded correlation coeficients in the 0.80 to 0.90 range, indicting good criterion-related validity. The 15 items are designed to tap behavioral domains, such as imitation, that have been reported as abnormal in autistic individuals. A four-point rating scale is used, with a score of one representing no abnormality and a score of four representing a severe abnormality. Thus, total CARS scores can range from 15 to 60. Scores of less than 30 are considered to be non-autistic, scores of 30 to 36 are considered to be mild to moderately autistic, and scores of 37 to 60 are considered to be severely autistic. The CARS is widely used to assist in establishing a diagnosis of autism and has now been standardized in a number of different countries, including Japan.*’ All the children described in this paper had a history of prenatal alcohol exposure and displayed the physical phenotype of FAS. However, the autistic FAS children failed to show the behavioral phenotype that has been described previously, but rather displayed the behavioral phenotype of autism. If autism does have multiple possible etiologies and is frequently associated with mental retardation, then it is reasonable to assume that prenatal exposure to teratogenic levels of alcohol can also serve as an etiologic agent for autism. SAMPLE
The age range represented in the data base is from newborns to young adults. Approximately 75% of the individuals with a primary diagnosis of FAS or ABRD in this data base are of North American Indian origin. This data base was reviewed for cases in which a secondary diagnosis of autism was recorded. The diagnosis of autism in this data base is made on the basis of the presence of abnormal behaviors and the natural history of the disorder. It is typically made after physical and psychological assessment. Scores on the Childhood Autism Rating Scale (CARS)*’ are used to standardize and quantify the clinical diagnosis. The CARS is administered by the psychologist during the assessment. A total of six cases with a diagnosis of both FAS and autism were found in the data base, four boys and two girls, aged 6 to 15 years. There were no cases of autism and ARBD. One male was of Caucasian origin, the remainder were of North American Indian origin. CASE REPORTS Case 1 This boy was born to a 34-year-old woman. He was the sixth child in the family. His mother was described by her primary care physician as a severe, chronic alcoholic who was unable to care adequately for the infant because of her alcohol abuse. Four of the older siblings were in foster care. At birth, he was noted to be small for gestational age and he thrived poorly. Extensive investigations for failure to thrive were negative. He was first noted to be mentally retarded at 2 years of age. At age 4 years, he was placed in foster care because ofpoor thriving and continuing alcohol abuse by his mother. At this time a diagnosis of FAS was made, based on facial dysmorphology, mental retardation, and delayed growth. Autistic behavior was also noted, consisting of ritualistic play, a dislike of body contact, abnormal fears and peculiar hand postures. This child has remained in the same foster home since then. At age five he had a prolonged hospitalization following a ruptured appendix. Following this, his behavior deteriorated, and a diagnosis of severe mental retardation and autism was made. At the time of this writing, he was 17 years of age and was attending a special class for retarded children. On the most recent assessment, he demonstrated significant self-abusive behavior; biting his hands and head-banging when upset. He was fascinated with objects that spin. Rapid mood swings without obvious stimuli was noted, and difficulty adapting to change in routines at home and at school were reported. He was also reported to have developed an insensitivity to pain. Both his foster mother and his teachers considered him to be hyperactive.
The Alvin Buckwold Centre is a multidisciplinary clinic for individuals with developmental disabilities. It serves as a tertiary referral center for mental retardation and other developmental disabilities in the province of Saskatche- Case 2 wan. A data base containing the records of 326 individuals This girl was born at 32 weeks following a motor vehicle accident in with FAS and other alcohol-related birth defects (ARBD) which her mother sustained mild injuries. She was the fourth child of a has been compiled over the past 10 years. Each subject 29-year-old mother. Her mother confirmed a history of alcohol abuse recorded in the data base has a history of maternal alcohol during the pregnancy. She reported drinking 12 to 24 bottles of beer on binges during the pregnancy. From ages 1 to 3 years, the child abuse during pregnancy, confirmed either by a drinking weekend was in foster care intermittently due to neglect and failure to thrive while history given by the biological mother or through social her parents underwent treatment for alcoholism. At age 3, she was service agency or medical records of alcohol abuse during permanently placed in a foster home, where she remains to this date. the pregnancy. All subjects also fulfill the diagnostic cri- Extensive investigations for failure to thrive were negative, and a diagteria for FAS or ABRD set out by RSA. This diagnosis is nosis of FAS was made based on facial dysmorphology, mild mental a fine tremor, and delayed growth. Autistic behaviors were based on a dysmorphology examination by a develop- retardation, first noted at the age of 3 years, and consisted of motor stereopathies mental pediatrician and assessment by a clinical psychol- such as self-spinning and turning objects over in front of her face. Her ogist. Other consultations such as cardiology are sought, speech was noted to be echolalic. This girl is now 14 years old and based on the dysmorphology examination and the history. attends a special class for retarded children. She is noted to be aloof with
’
56 1
AUTISM IN FETAL ALCOHOL SYNDROME
strangers but affectionate with her foster family. She is obsessed with purses, carrying one everywhere. She has ritualistic play in which she twirls her fingers in front of her face. She is able to carry a conversation but her range of topics is limited. She has some limited reading skills but has great difficulty with math concepts such as time and money. She is reported to be insensitive to pain and not aware of dangers. Case 3
This boy was the seventh child of a 29-year-old mother. He had a ventral septal defect, cleft palate, bilateral metatarsus varus, and inguinal hernia, that necessitated his transfer to a large medical center. Following successful surgeries to correct these defects, he was placed in a foster home at 4 months of age where he has remained to date. He has intermittent contact with his natural family. At age 2 he was noted to be below the third percentile for weight, height, and head circumference, to have facial dysmorphology, mental retardation, and mild spastic cerebral palsy. Contact with his mother’s primary care physician confirmed a history of alcohol abuse during the pregnancy, and a diagnosis of FAS was made. Estimates of his mother’s consumption patterns were unavailable. At the age of 6 years, he was noted to spin objects such as the wheels of toy cars, to play ritualistically, and to resist change. A diagnosis of autism was made at that time. He is now 14 years old and attends a special school for retarded children; he receives assistance from a recreational program for autistic individuals. He has only a few words of speech and is not toilet trained. He shows great difficulty adapting to changes in routines, including changes in the texture or temperature of foods. He cries easily if upset and is difficult to comfort. This foster mother describes his activity level as ‘‘lethargic.’’ Case 4
This boy was adopted at 3 months of age, as an apparently normal child. His adoptive parents were informed that the mother had been a healthy teenager, of Caucasian origin, who had drunk on weekend “binges.” No estimates of the quantities of alcohol consumed were available. He thrived poorly and was slow reaching all developmental milestones. Investigation for failure to thrive was negative, and at age 4 a diagnosis of fetal alcohol syndrome was made, based on facial dysmorphology, severe growth retardation, and mental retardation. A concurrent diagnosis of autism was also made, based on the DSM 111 criteria“ for this disorder. He is now 11 years old and attends a special class in a regular school. He is described as hyperactive, fearless, having no awareness of danger, and having a very high pain tolerance. He is fascinated with light switches and will flick these for long periods of time. Speech is limited to sentences of less than five words and consists mostly of echolalia. Case 5
This girl is the fifth child and was born at term to a 28-year-old woman. The four older children are all in foster care. There was a history of severe alcohol abuse during the fifth pregnancy, with multiple hospital admissions during the pregnancy for alcohol gastritis. At birth, the baby was noted to have a left diaphragmatic hernia, cleft lip and palate, a ventral septal defect, and mitral valve prolapse. The child was hospitalized for the first 9 months of life and had multiple surgeries. Her mother cared for her briefly and then placed her in a foster home where she was subsequently adopted. Some contact with her biological mother is maintained. She has one younger brother with FAS, who is mildly retarded and hyperactive, who lives with his mother. At 1 year of age she was diagnosed as FAS and was assessed as being mentally retarded. Autistic behaviors were first noted at age 4. She is now 7 years of age and functions in the severely retarded range. She has no speech and is tutored at home as severe allergies and asthma prevent her from being in a larger group setting. She shows a number of self-stimulating behaviors such as rocking, masturbation, and flicking her fingers in front of her face. She
is easily upset by change, especially when new foods are introduced. She appears hypersensitive to sounds. She has no speech but uses a form of sign language to communicate basic needs. Case 6
This boy is the youngest of three children, born to a 25-year-old woman. There was a history of alcohol abuse and glue sniffing during this pregnancy. No estimates of quantity were available. There has been no contact between this child and his biological mother for many years. He was diagnosed at birth as FAS, based on the presence of growth retardation, facial dysmorphology, and a tremor. A cleft of the soft palate was noted at birth. An older sister is reported to have FAS, and to be severely retarded but not autistic. This boy has remained in the same foster home since 6 days of age. Mild cerebral palsy and hyperactivity have been diagnosed in addition to FAS and autism. He demonstrated islands of intelligence such as knowing all the color names but was unable to use this skill consistently. He demonstrates ritualistic play, consisting of lining up toys in sequences. He avoids eye contact and is upset by changes in routine. He has pica and will eat non-nutritive substances if not watched. He also has no sense of danger.
COMPARISON WITH OTHER FAS CHILDREN2
In order to assess whether these six children were different from the other FAS individuals followed by this clinic in terms of their history or behavior, the data base was reviewed for FAS children within the same age range (516 years) with similar levels of intelligence (IQs 20-55), without a diagnosis of autism or any record of autistic behavior. Children who were considered untestable due to sensory or physical handicaps or with ARBD but not FAS were excluded. A further eight children fulfilled all of these criteria. There were six girls and two boys in this group. All were of North American Indian origin. These children had all been previously examined by the same pediatrician as had examined the autistic FAS children. The records of these eight contrasting children were examined for details of their physical development and social history to determine between group (autistic and non-autistic FAS) differences. Physical variables included gestational age, birth weight, growth indices, and frequency of major malformations. Growth indices were obtained from the most recent assessment at the Alvin Buckwold Centre. As growth charts for Canadian Indian children are not available, standard growth charts were used,29as have similar studies on FAS children of Indian origin6 Typically multiple measures of growth were available. The most recent measurements were used for the analyses. Measurements were transformed into standard deviation scores for the analyses. Social variables at the time of the most recent assessment included current home status (fostered or adopted), total number of foster or adoptive home placements,and school placement (regular or special class). Between group IQ scores based on the most recent IQ data available were compared. All subjects had been assessed using either the Wechsler Intelligence Scale for Children-Re~ised~’ or in the case of lower functioning children, the Stanford Binet scale^.^'.^^ Because CARS scores were not available for the
562
NANSON
non-autistic FAS contrast group, their caregivers were contacted and interviewed in regard to autistic symptoms. None of these individuals showed significant autism, although all had behavior problems by caregiver report. RESULTS
The results of the comparison between the autistic FAS children and the non-autistic, FAS contrast group are presented in Table 1. The autistic FAS children were significantly older ( 131 months) than the non-autistic FAS children (106 months) at the time of the most recent assessment. There were no differences in the ages at which FAS had been diagnosed (31.8 vs. 28.8 months). The mean age at which autism had been diagnosed was 73 months. There were no significant differences between the autistic and non-autistic groups in terms of birth weights or gestational ages. However, the statistical analyses were based on very small sample sizes, as gestational ages were known for only 12 of the children. The autistic FAS group demonstrated a trend toward greater growth retardation at the time of most recent examination (height, weight, and head circumference) than did the children in the nonautistic FAS group, although this did not reach statistical significance. However, the autistic FAS children were significantly older than the non-autistic FAS children, in spite of attempts to match for age. The fact that the autistic FAS children were similarly sized or slightly smaller than the non-autistic FAS children at an older age suggests that the autistic FAS children did experience greater growth retardation than did the non-autistic FAS children. A number of the children (particularly the non-autistic FAS ones) had experienced “catch-up” growth, particularly in weight, since their initial measurements. This “catch-up” growth in weight has been reported in other studies6 There were no differences between the two groups in terms of the number of children with major malformations present. However, the autistic FAS children had a greater absolute number of anomalies present (Table 2). In addition, three of the six autistic FAS children had cleft lip and/or palate. Although cleft lip and/or palate has been previously reported in cases of FAS,’ the incidence in this Table 1. Growth of Children with FAS and Autism as Compared with Children with Only FAS
Gestationalage < 37 wks
FAS/ Autistic
FAS
Statistic
p
2 (5)
0(8)
Chi Square
NS’
2492 (6) 1361-2948
2864 (6) 1800-3640
t
NS
131 -1.29 -1.34 -3.25
106 -2.03 -0.63 -2.10
t
0.001 NS NS NS
(N) Birth weight (9) (N) (range) Age at most recent Assessment (months) Mean height (SD units) Mean weight (SD units) Mean head circumference (SD units) * NS, not significant.
t t t
sample is very high. Cleft lip and/or palate is not commonly associated with the diagnosis of There were no statistically significant differences between the two groups in terms of the current living arrangements (Table 3). Two of the children in each group had been adopted, and one non-autistic FAS girl continued to live with her biological parents. The remainder were in long-term, foster placements. One autistic FAS girl had been in several temporary placements being permanently placed at age 3 years. All the other children had experienced three or fewer placements. All but one of these children had been placed in foster care prior to age 4, either because of abuse and neglect associated with parental alcohol abuse or because of their fragile physical health. The exception was one autistic child who had been given up for adoption at birth. Two autistic and two nonautistic children had contact with their biological mothers as well. Four of the non-autistic FAS children were placed in a mainstream classroom, although not necessarily with age peers. All of the autistic FAS children were in special classes or schools for the retarded except one who was tutored at home due to poor health. In terms of intellectual functioning, the non-autistic children were significantly less retarded (mean IQ 47) than were the autistic FAS children (mean IQ 33). The autistic FAS children all functioned in the severely autistic range, according to their CARS scores. The autistic FAS children were described as having many of the characteristics of autism that Schreibman” suggests do not overlap with mental retardation. These included difficulty relating to people and resistance to change in daily routines, they also showed abnormal sensory responses such as high pain thresholds or hypersensitivity to noise. Only two autistic children used speech consistently and both showed the echolalia and pronoun reversal, that are characteristic of autistic language use.21All of the non-autistic FAS children had language and none had a history of echolalia or pronominal reversal. Thus, the autistic FAS children tended to show the behavioral features of autism that are not characteristic of all severely retarded individuals. All of these children, regardless of other diagnoses, were described by their caregivers as lacking in socially appropriate and age-appropriate fears, such as a fear of strangers or of physical danger. All 14 children were noted to have some unusual movements such as clumsiness, rocking, or head banging, although these were generally more severe in the autistic children. Finally, all but one of the children were judged by their caregivers to be over-active when compared with other children. The remaining autistic FAS boy with mild cerebral palsy was rated by his foster mother as “sluggish.” DISCUSSION
Autism is not usually included in the range of bchavior problems known to be associated with FAS, although the
AUTISM IN FETAL ALCOHOL SYNDROME
563 Table 2. Patient Characteristics
Patient No. Age at Assessment (months)
Diagnosis
1 (184) 2 (170) 3 (171)
Sex
Gestational Age (weeks)
Birth Weight (9)
FAS/Autisrn FAS/Autism FAS/Autism
M F M
34 32 38
2320 1361 2570
4 (128)
FAS/Autism
M
Unknown
2940
5 (82)
FAS/Autism
F
40
2890
6 (68)
FAS/Autism
M
40
7 (161) 8 (120) 9 (55)
FAS FAS FAS
F F M
10 (138) 12 (66) 13 (88)
FAS FAS FAS FAS
F M F
14 (169)
FAS
11 (52)
Major Malformations
CNS Dysfunction
IQ
CARS
Ht
Wt
HC
(SDunits) None None Ventral septal defect Cleft palate Metatarsus varus Inguinal hernia None
Mental retardation Mental retardation Mental retardation Spastic cerebral Palsy
28 54 30
52 38 51
-2.07 -1.68 -3.15
-2.30 -1.05 -0.37
-3.93 -5.12 -2.34
Mental retardation
20 30
44 46
-2.32
-0.94
-1.81
Mental retardation
-2.24
-1.86
-3.59
2850
Ventral septal defect Mitral valve prolapse Cleft lip and palate Diaphramatic hernia Cleft lip and palate
-0.70
-1.57
-2.71
Unknown Unknown 40
1800 Unknown 2760
None Ventral septal defect Tetralogy of fallot
43 39 53
-3.97 -1.18 -0.49
-0.96 0.78 -0.46
-2.75 0.88 -0.63
F
40 40 40 40
1818 2640 2990 3440
Ventral septal defect None None Ventral septal defect
53 54 54 50
-1.38 -2.70 -3.75 -0.46
-0.67 -2.58 -2.30 -0.12
-2.74 -6.43 -3.75 -0.08
F
Unknown
Unknown
None
Table 3. Current Status of Children with FAS and Autism as Compared with Children with Only FAS
Mental retardation Cerebral palsy Hyperactivity Mental retardation Mental retardation Mental retardation Hyperactivity Mental retardation Mental retardation Mental retardation Mental retardation Spastic cerebral palsy Mental retardation Seizures
35
30
46
1.64
1.20
-4.00
children may have suffered greater prenatal insult than did the non-autistic children. Estimates of maternal alcoFAS/ hol intake during pregnancy were not available so that it Autistic FAS Statistic P was not possible to determine whether the autistic FAS 2 (6) 2 (8) Chi Square NS Adopted ( N ) 1 (6) 0 (8) Chi Square NS Number of foster children had been exposed to greater amounts of alcohol placements >3? in utero. Prenatal alcohol exposure has not previously 6 (6) 4 (8) Chi Square
Vol. 16, No. 3 May/June 1992
Autism in Fetal Alcohol Syndrome: A Report of Six Cases J. L. Nanson
This paper reports on six children who all fulfill the criteria for fetal alcohol syndrome and have a history of maternal alcohol abuse during pregnancy, and who also fulfill the criteria for diagnosis of autism. Their behavior and natural history is compared with eight contrast FAS children, of similar ages and functional levels, who were free of autistic behaviors. The autistic FAS children were all moderately or severely retarded. Autistic behavior has not been commonly associated with prenatal alcohol exposure. Clinicians need to be aware of this potentialdual diagnosis so that appropriate educational and treatment resources can be employed. Key Words: Fetal Alcohol Syndrome, Physical Growth, Autism, Mental Retardation.
RENATAL EXPOSURE to excessive maternal alcoP hol ingestion results in a wide variety of defects ranging from life threatening anomalies to subtle problems with learning and attention. Although the variability in physical manifestations of alcohol-related birth defects is well recognized, variability in the behavioral manifestations of prenatal alcohol exposure is less well described in the literature. The purpose of this paper is to describe six children, ranging in age from 6 to 15 years, who fulfill the criteria for a diagnosis of both fetal alcohol syndrome (FAS) and autism. The behavior of these children is very different from previously published descriptions of behavior in children with FAS, particularly in regard to their social and emotional f~ n c t i o n i n g . ~ In-~order to assess the extent to which these children are different from other FAS children, their behavior and social histories are compared with a contrast group of FAS children, matched for age and degree of retardation who did not show the characteristics of autism. The diagnostic criteria for FAS have been revised by the Fetal Alcohol Study Group of the Research Society on Alcoholism (RSA)’ to include prenatal and/or postnatal growth retardation of weight and height, craniofacial anomalies, and evidence of central nervous system dysfunction such as developmental delay, mental retardation, hyperactivity, or tremor. In the literature, reported cases of FAS have varied greatly in terms of the degree of cognitive impairment present, from IQs within the normal range to IQs in the severely retarded range, with mean IQs typically in the mildly retarded or borderline ranges.8 Streissguth and Ladue’ have suggested that there is a ‘7’
From the Alvin Buckwold Centre, Royal University Hospital, Saskatoon, SK, Canada. Received for publication June 20, 1990; accepted December 3, I991 Reprint requests: Dr. JL Nanson, Alvin Buckwold Centre, Royal University Hospital, Saskatoon, SK Canada S7N 0 WO. Copyright 0 1992 by The Research Society on Alcoholism. 558
behavioral phenotype of FAS, with unique manifestations in different developmental levels. In young children, impulsivity, hyperactivity, over-friendliness, poor social judgement, and a sprightly, butterfly-like manner or movement were noted. In school age children, hyperactivity continued to be problematic and to account for some of their significant learning difficulty in school. The authors describe the highly social orientation of these children, that often gives the impression that they are more intelligent than intelligence test scores suggest. Problems with over-talkativeness, over-intrusive behavior, and poor social judgement persist into adolescence and adulthood, and contribute to significant loneliness and few friendships. The children described in this paper manifested the physical phenotype of FAS, and had a history of maternal alcohol abuse during pregnancy but had a very different behavioral phenotype, typical of autistic children, and characterized by more significant retardation of both their cognitive and social skills. In contrast to FAS, which is diagnosed on the basis of growth retardation, dysmorphology and central nervous system (CNS) dysfunction, and is associated with a uniform etiology, autism is diagnosed on the basis of the child’s behavior and the natural history of the disorder, without reference to growth, dysmorphology, or etiology. Although the incidence of dysmorphic features is increased in the autistic population relative to normal individuals, no isolated defect or pattern of dysmorphic features has been reliably associated with autism.’ Similarly, recent work” has failed to isolate a single factor or combination of factors that contributed to the etiology of autism. Although a psychogenic etiology of autism was initially suggested when Kanner” first described the disorder in 1943, subsequent research has failed to support this idea. Current research in the area is primarily focused on neurobiological aspects of the disorder now.” A neurobiological basis for autism is suggested by the increased incidence within some families” and the increased frequency of mental retardation and other symptoms of central nervous system impairment in individuals with autism. Kanner’ I originally thought that autistic individuals were of normal intelligence but often had low scores on intelligence tests because of their defects in social relationships. Subsequent research has failed to support this view, and it is now thought that the majority of autistic individuals are also mentally retarded. For example, Ritvo and FreemanI3 have suggested that 60% of autistic children have IQs below 50, 20% have IQs between 50 and 70, and only Alcohol Clin Exp Res. Vol 16, No 3 , 1992: pp 558-565
AUTISM IN FETAL ALCOHOL SYNDROME
20% have IQs above 70. Thus, mental retardation and autism are separate disorders but frequently co-exist in the same individual. Autistic individuals are also more likely to have uneven intellectual profiles whereas nonautistic, retarded individuals are more likely to have uniform defects in all areas as~essed.’~ Coleman” has suggested that autism is a syndrome with many different etiologies. Chromosomal disorders, such as fragile X; prenatal infections, especially rubella; untreated metabolic conditions, such as phenylketonuria; and other genetic syndromes, such as tuberous sclerosis are also associated with an increased incidence of autism. In almost all of these conditions, the affected individuals are also mentally retarded. The hallmark of autism is a defect in social behavior and attachment. Other defects include problems with both verbal and nonverbal communication, and perseverative, stereotypic and ritualistic behaviors. In spite of general agreement on the core features of the disorder, the diagnostic criteria for the disorder are still a matter of some dispute amongst clinicians. In the Revised Diagnostic and Statistic Manual of Mental Disorders (DSM-I 1 1R),I6autism is classified as one of several disorders originating in infancy and early childhood subsumed under the title of Pervasive Developmental Disorders (PDD). The diagnostic criteria for PDD include both behavioral and historical components. Allen” has argued that all of the pervasive developmental disorders listed in the DSM-I 11R are variants of autism and should be classified as such. She has suggested that the variability in the clinical presentation of autism is a consequence of qualitative differences in social, language, play, and communication skills. SchreibmanI8 has reviewed the behavioral features of autism and listed eight key deficits that clinicians typically use in making the diagnosis of autism. Of these, six are much more common in autism than other disorders, while the remaining four overlap with mental retardation. The six unique features are: I. A defect in social behavior. Autistic children, regardless of their intellectual abilities, appear detached from their social environment. They prefer to be alone, and may ignore or react negatively to affection and social interactions offered by parents and other children. 2. Defects in speech and language. Approximately half of all autistic individuals never develop any meaningful speech,”-” and those that do typically have language use that is qualitatively different from other children. Echolalia (repetition of words and phrases spoken by others), and pronominal reversal (using you for I and proper names for me), as well as defects in comprehension and speech rhythms (prosody) are also character is ti^.^^-*^ 3. The preservation of sameness. This was first identified by Kanner” as a key feature of autism. Autistic individuals are typically very sensitive to minor changes in their environment, such as a change in the location of a toy or
559
a new activity added to daily routines. Often associated with this need for sameness is a ritualistic preoccupation with a single activity such as memorizing maps or train schedules. Ritualistic eating patterns, such as consuming only one food, are also common. 4. Abnormal responses to their sensory environment. Both under- and over-responsiveness to sensory stimulation are reported.25Often these individuals are suspected of being deaf because of their lack of responsiveness to sounds but on examination, have normal hearing. The same individual may show extreme reactions to certain classes of sounds. Similar, variable, and inconsistent responses to taste, touch, and pain are also common.*’ 5 . Exceptional abilities in one isolated area such as music or mathematics.26This skill may represent one area of average functioning in an individual with depressed functioning in a number of other areas or may represent a truly exceptional ability. 6. Emotional responsiveness or affect. This final area is usually identified as abnormal in autistic individuals, regardless of their intellectual abilities. Autistic individuals may show flattening of affect, or have rapid and unpredictable mood swings that appear unrelated to social events. The remaining diagnostic features: self-stimulatory behaviors, such as rocking, hand flapping, and spinning and self-injurious behavior, such as head banging or biting of hands are characteristic of autistic individuals,” but are also common in the mentally retarded, especially those with sensory impairments, such as the deaf-blindI4and in girls with Rett syndrome.25Self-stimulatory behaviors may preoccupy an autistic individual to the preclusion of all other activities. These behaviors are often resistant to treatment. The diagnosis of autism is typically made between the age of 2 to 4 years of age when the affected child fails to develop appropriate speech and social skills. Both the natural history of the disorder and the child’s behavior are used in making the diagnosis. Although a number of abnormalities have been reported as more frequent in autism, there is no single investigation or finding that will confirm a diagnosis of autism. Rather it is the presence of the constellation of symptoms that leads to a diagnosis. Most clinicians do not view autism as a well-defined syndrome with a single etiology but rather as a spectrum of behaviors, which can characterize individuals with very different levels of intelligence. In order to assist with the differentiation of this spectrum of autistic behaviors, a number of different types of rating scales have been developed. One of the more recent of these is the Childhood Autism Rating Scale.27The CARS is a 15-item scale that is designed to differentiate between autistic and non-autistic children, with and without concomitant mental retardation. It is designed to be administered by a psychologist, using the parent or someone else familiar with the child as an informant. The items
560
NANSON
are based on five diagnostic systems that have been proposed for the diagnosis of autism, including the DSM I11 criteria. The items are similar to those described by Schreibman (see above). The CARS is based on studies involving over 1500 children of all ages, both retarded and non-retarded who have been assessed by Schopler and his colleague^^^ in their program for autistic children at the University of North Carolina. Internal consistency, interrater and test reliability are all reported as with acceptable limits for a behavioral rating scale.The validity of this scale was assessed by correlating CARS scores with independent clinical diagnoses of autism. Different samples yielded correlation coeficients in the 0.80 to 0.90 range, indicting good criterion-related validity. The 15 items are designed to tap behavioral domains, such as imitation, that have been reported as abnormal in autistic individuals. A four-point rating scale is used, with a score of one representing no abnormality and a score of four representing a severe abnormality. Thus, total CARS scores can range from 15 to 60. Scores of less than 30 are considered to be non-autistic, scores of 30 to 36 are considered to be mild to moderately autistic, and scores of 37 to 60 are considered to be severely autistic. The CARS is widely used to assist in establishing a diagnosis of autism and has now been standardized in a number of different countries, including Japan.*’ All the children described in this paper had a history of prenatal alcohol exposure and displayed the physical phenotype of FAS. However, the autistic FAS children failed to show the behavioral phenotype that has been described previously, but rather displayed the behavioral phenotype of autism. If autism does have multiple possible etiologies and is frequently associated with mental retardation, then it is reasonable to assume that prenatal exposure to teratogenic levels of alcohol can also serve as an etiologic agent for autism. SAMPLE
The age range represented in the data base is from newborns to young adults. Approximately 75% of the individuals with a primary diagnosis of FAS or ABRD in this data base are of North American Indian origin. This data base was reviewed for cases in which a secondary diagnosis of autism was recorded. The diagnosis of autism in this data base is made on the basis of the presence of abnormal behaviors and the natural history of the disorder. It is typically made after physical and psychological assessment. Scores on the Childhood Autism Rating Scale (CARS)*’ are used to standardize and quantify the clinical diagnosis. The CARS is administered by the psychologist during the assessment. A total of six cases with a diagnosis of both FAS and autism were found in the data base, four boys and two girls, aged 6 to 15 years. There were no cases of autism and ARBD. One male was of Caucasian origin, the remainder were of North American Indian origin. CASE REPORTS Case 1 This boy was born to a 34-year-old woman. He was the sixth child in the family. His mother was described by her primary care physician as a severe, chronic alcoholic who was unable to care adequately for the infant because of her alcohol abuse. Four of the older siblings were in foster care. At birth, he was noted to be small for gestational age and he thrived poorly. Extensive investigations for failure to thrive were negative. He was first noted to be mentally retarded at 2 years of age. At age 4 years, he was placed in foster care because ofpoor thriving and continuing alcohol abuse by his mother. At this time a diagnosis of FAS was made, based on facial dysmorphology, mental retardation, and delayed growth. Autistic behavior was also noted, consisting of ritualistic play, a dislike of body contact, abnormal fears and peculiar hand postures. This child has remained in the same foster home since then. At age five he had a prolonged hospitalization following a ruptured appendix. Following this, his behavior deteriorated, and a diagnosis of severe mental retardation and autism was made. At the time of this writing, he was 17 years of age and was attending a special class for retarded children. On the most recent assessment, he demonstrated significant self-abusive behavior; biting his hands and head-banging when upset. He was fascinated with objects that spin. Rapid mood swings without obvious stimuli was noted, and difficulty adapting to change in routines at home and at school were reported. He was also reported to have developed an insensitivity to pain. Both his foster mother and his teachers considered him to be hyperactive.
The Alvin Buckwold Centre is a multidisciplinary clinic for individuals with developmental disabilities. It serves as a tertiary referral center for mental retardation and other developmental disabilities in the province of Saskatche- Case 2 wan. A data base containing the records of 326 individuals This girl was born at 32 weeks following a motor vehicle accident in with FAS and other alcohol-related birth defects (ARBD) which her mother sustained mild injuries. She was the fourth child of a has been compiled over the past 10 years. Each subject 29-year-old mother. Her mother confirmed a history of alcohol abuse recorded in the data base has a history of maternal alcohol during the pregnancy. She reported drinking 12 to 24 bottles of beer on binges during the pregnancy. From ages 1 to 3 years, the child abuse during pregnancy, confirmed either by a drinking weekend was in foster care intermittently due to neglect and failure to thrive while history given by the biological mother or through social her parents underwent treatment for alcoholism. At age 3, she was service agency or medical records of alcohol abuse during permanently placed in a foster home, where she remains to this date. the pregnancy. All subjects also fulfill the diagnostic cri- Extensive investigations for failure to thrive were negative, and a diagteria for FAS or ABRD set out by RSA. This diagnosis is nosis of FAS was made based on facial dysmorphology, mild mental a fine tremor, and delayed growth. Autistic behaviors were based on a dysmorphology examination by a develop- retardation, first noted at the age of 3 years, and consisted of motor stereopathies mental pediatrician and assessment by a clinical psychol- such as self-spinning and turning objects over in front of her face. Her ogist. Other consultations such as cardiology are sought, speech was noted to be echolalic. This girl is now 14 years old and based on the dysmorphology examination and the history. attends a special class for retarded children. She is noted to be aloof with
’
56 1
AUTISM IN FETAL ALCOHOL SYNDROME
strangers but affectionate with her foster family. She is obsessed with purses, carrying one everywhere. She has ritualistic play in which she twirls her fingers in front of her face. She is able to carry a conversation but her range of topics is limited. She has some limited reading skills but has great difficulty with math concepts such as time and money. She is reported to be insensitive to pain and not aware of dangers. Case 3
This boy was the seventh child of a 29-year-old mother. He had a ventral septal defect, cleft palate, bilateral metatarsus varus, and inguinal hernia, that necessitated his transfer to a large medical center. Following successful surgeries to correct these defects, he was placed in a foster home at 4 months of age where he has remained to date. He has intermittent contact with his natural family. At age 2 he was noted to be below the third percentile for weight, height, and head circumference, to have facial dysmorphology, mental retardation, and mild spastic cerebral palsy. Contact with his mother’s primary care physician confirmed a history of alcohol abuse during the pregnancy, and a diagnosis of FAS was made. Estimates of his mother’s consumption patterns were unavailable. At the age of 6 years, he was noted to spin objects such as the wheels of toy cars, to play ritualistically, and to resist change. A diagnosis of autism was made at that time. He is now 14 years old and attends a special school for retarded children; he receives assistance from a recreational program for autistic individuals. He has only a few words of speech and is not toilet trained. He shows great difficulty adapting to changes in routines, including changes in the texture or temperature of foods. He cries easily if upset and is difficult to comfort. This foster mother describes his activity level as ‘‘lethargic.’’ Case 4
This boy was adopted at 3 months of age, as an apparently normal child. His adoptive parents were informed that the mother had been a healthy teenager, of Caucasian origin, who had drunk on weekend “binges.” No estimates of the quantities of alcohol consumed were available. He thrived poorly and was slow reaching all developmental milestones. Investigation for failure to thrive was negative, and at age 4 a diagnosis of fetal alcohol syndrome was made, based on facial dysmorphology, severe growth retardation, and mental retardation. A concurrent diagnosis of autism was also made, based on the DSM 111 criteria“ for this disorder. He is now 11 years old and attends a special class in a regular school. He is described as hyperactive, fearless, having no awareness of danger, and having a very high pain tolerance. He is fascinated with light switches and will flick these for long periods of time. Speech is limited to sentences of less than five words and consists mostly of echolalia. Case 5
This girl is the fifth child and was born at term to a 28-year-old woman. The four older children are all in foster care. There was a history of severe alcohol abuse during the fifth pregnancy, with multiple hospital admissions during the pregnancy for alcohol gastritis. At birth, the baby was noted to have a left diaphragmatic hernia, cleft lip and palate, a ventral septal defect, and mitral valve prolapse. The child was hospitalized for the first 9 months of life and had multiple surgeries. Her mother cared for her briefly and then placed her in a foster home where she was subsequently adopted. Some contact with her biological mother is maintained. She has one younger brother with FAS, who is mildly retarded and hyperactive, who lives with his mother. At 1 year of age she was diagnosed as FAS and was assessed as being mentally retarded. Autistic behaviors were first noted at age 4. She is now 7 years of age and functions in the severely retarded range. She has no speech and is tutored at home as severe allergies and asthma prevent her from being in a larger group setting. She shows a number of self-stimulating behaviors such as rocking, masturbation, and flicking her fingers in front of her face. She
is easily upset by change, especially when new foods are introduced. She appears hypersensitive to sounds. She has no speech but uses a form of sign language to communicate basic needs. Case 6
This boy is the youngest of three children, born to a 25-year-old woman. There was a history of alcohol abuse and glue sniffing during this pregnancy. No estimates of quantity were available. There has been no contact between this child and his biological mother for many years. He was diagnosed at birth as FAS, based on the presence of growth retardation, facial dysmorphology, and a tremor. A cleft of the soft palate was noted at birth. An older sister is reported to have FAS, and to be severely retarded but not autistic. This boy has remained in the same foster home since 6 days of age. Mild cerebral palsy and hyperactivity have been diagnosed in addition to FAS and autism. He demonstrated islands of intelligence such as knowing all the color names but was unable to use this skill consistently. He demonstrates ritualistic play, consisting of lining up toys in sequences. He avoids eye contact and is upset by changes in routine. He has pica and will eat non-nutritive substances if not watched. He also has no sense of danger.
COMPARISON WITH OTHER FAS CHILDREN2
In order to assess whether these six children were different from the other FAS individuals followed by this clinic in terms of their history or behavior, the data base was reviewed for FAS children within the same age range (516 years) with similar levels of intelligence (IQs 20-55), without a diagnosis of autism or any record of autistic behavior. Children who were considered untestable due to sensory or physical handicaps or with ARBD but not FAS were excluded. A further eight children fulfilled all of these criteria. There were six girls and two boys in this group. All were of North American Indian origin. These children had all been previously examined by the same pediatrician as had examined the autistic FAS children. The records of these eight contrasting children were examined for details of their physical development and social history to determine between group (autistic and non-autistic FAS) differences. Physical variables included gestational age, birth weight, growth indices, and frequency of major malformations. Growth indices were obtained from the most recent assessment at the Alvin Buckwold Centre. As growth charts for Canadian Indian children are not available, standard growth charts were used,29as have similar studies on FAS children of Indian origin6 Typically multiple measures of growth were available. The most recent measurements were used for the analyses. Measurements were transformed into standard deviation scores for the analyses. Social variables at the time of the most recent assessment included current home status (fostered or adopted), total number of foster or adoptive home placements,and school placement (regular or special class). Between group IQ scores based on the most recent IQ data available were compared. All subjects had been assessed using either the Wechsler Intelligence Scale for Children-Re~ised~’ or in the case of lower functioning children, the Stanford Binet scale^.^'.^^ Because CARS scores were not available for the
562
NANSON
non-autistic FAS contrast group, their caregivers were contacted and interviewed in regard to autistic symptoms. None of these individuals showed significant autism, although all had behavior problems by caregiver report. RESULTS
The results of the comparison between the autistic FAS children and the non-autistic, FAS contrast group are presented in Table 1. The autistic FAS children were significantly older ( 131 months) than the non-autistic FAS children (106 months) at the time of the most recent assessment. There were no differences in the ages at which FAS had been diagnosed (31.8 vs. 28.8 months). The mean age at which autism had been diagnosed was 73 months. There were no significant differences between the autistic and non-autistic groups in terms of birth weights or gestational ages. However, the statistical analyses were based on very small sample sizes, as gestational ages were known for only 12 of the children. The autistic FAS group demonstrated a trend toward greater growth retardation at the time of most recent examination (height, weight, and head circumference) than did the children in the nonautistic FAS group, although this did not reach statistical significance. However, the autistic FAS children were significantly older than the non-autistic FAS children, in spite of attempts to match for age. The fact that the autistic FAS children were similarly sized or slightly smaller than the non-autistic FAS children at an older age suggests that the autistic FAS children did experience greater growth retardation than did the non-autistic FAS children. A number of the children (particularly the non-autistic FAS ones) had experienced “catch-up” growth, particularly in weight, since their initial measurements. This “catch-up” growth in weight has been reported in other studies6 There were no differences between the two groups in terms of the number of children with major malformations present. However, the autistic FAS children had a greater absolute number of anomalies present (Table 2). In addition, three of the six autistic FAS children had cleft lip and/or palate. Although cleft lip and/or palate has been previously reported in cases of FAS,’ the incidence in this Table 1. Growth of Children with FAS and Autism as Compared with Children with Only FAS
Gestationalage < 37 wks
FAS/ Autistic
FAS
Statistic
p
2 (5)
0(8)
Chi Square
NS’
2492 (6) 1361-2948
2864 (6) 1800-3640
t
NS
131 -1.29 -1.34 -3.25
106 -2.03 -0.63 -2.10
t
0.001 NS NS NS
(N) Birth weight (9) (N) (range) Age at most recent Assessment (months) Mean height (SD units) Mean weight (SD units) Mean head circumference (SD units) * NS, not significant.
t t t
sample is very high. Cleft lip and/or palate is not commonly associated with the diagnosis of There were no statistically significant differences between the two groups in terms of the current living arrangements (Table 3). Two of the children in each group had been adopted, and one non-autistic FAS girl continued to live with her biological parents. The remainder were in long-term, foster placements. One autistic FAS girl had been in several temporary placements being permanently placed at age 3 years. All the other children had experienced three or fewer placements. All but one of these children had been placed in foster care prior to age 4, either because of abuse and neglect associated with parental alcohol abuse or because of their fragile physical health. The exception was one autistic child who had been given up for adoption at birth. Two autistic and two nonautistic children had contact with their biological mothers as well. Four of the non-autistic FAS children were placed in a mainstream classroom, although not necessarily with age peers. All of the autistic FAS children were in special classes or schools for the retarded except one who was tutored at home due to poor health. In terms of intellectual functioning, the non-autistic children were significantly less retarded (mean IQ 47) than were the autistic FAS children (mean IQ 33). The autistic FAS children all functioned in the severely autistic range, according to their CARS scores. The autistic FAS children were described as having many of the characteristics of autism that Schreibman” suggests do not overlap with mental retardation. These included difficulty relating to people and resistance to change in daily routines, they also showed abnormal sensory responses such as high pain thresholds or hypersensitivity to noise. Only two autistic children used speech consistently and both showed the echolalia and pronoun reversal, that are characteristic of autistic language use.21All of the non-autistic FAS children had language and none had a history of echolalia or pronominal reversal. Thus, the autistic FAS children tended to show the behavioral features of autism that are not characteristic of all severely retarded individuals. All of these children, regardless of other diagnoses, were described by their caregivers as lacking in socially appropriate and age-appropriate fears, such as a fear of strangers or of physical danger. All 14 children were noted to have some unusual movements such as clumsiness, rocking, or head banging, although these were generally more severe in the autistic children. Finally, all but one of the children were judged by their caregivers to be over-active when compared with other children. The remaining autistic FAS boy with mild cerebral palsy was rated by his foster mother as “sluggish.” DISCUSSION
Autism is not usually included in the range of bchavior problems known to be associated with FAS, although the
AUTISM IN FETAL ALCOHOL SYNDROME
563 Table 2. Patient Characteristics
Patient No. Age at Assessment (months)
Diagnosis
1 (184) 2 (170) 3 (171)
Sex
Gestational Age (weeks)
Birth Weight (9)
FAS/Autisrn FAS/Autism FAS/Autism
M F M
34 32 38
2320 1361 2570
4 (128)
FAS/Autism
M
Unknown
2940
5 (82)
FAS/Autism
F
40
2890
6 (68)
FAS/Autism
M
40
7 (161) 8 (120) 9 (55)
FAS FAS FAS
F F M
10 (138) 12 (66) 13 (88)
FAS FAS FAS FAS
F M F
14 (169)
FAS
11 (52)
Major Malformations
CNS Dysfunction
IQ
CARS
Ht
Wt
HC
(SDunits) None None Ventral septal defect Cleft palate Metatarsus varus Inguinal hernia None
Mental retardation Mental retardation Mental retardation Spastic cerebral Palsy
28 54 30
52 38 51
-2.07 -1.68 -3.15
-2.30 -1.05 -0.37
-3.93 -5.12 -2.34
Mental retardation
20 30
44 46
-2.32
-0.94
-1.81
Mental retardation
-2.24
-1.86
-3.59
2850
Ventral septal defect Mitral valve prolapse Cleft lip and palate Diaphramatic hernia Cleft lip and palate
-0.70
-1.57
-2.71
Unknown Unknown 40
1800 Unknown 2760
None Ventral septal defect Tetralogy of fallot
43 39 53
-3.97 -1.18 -0.49
-0.96 0.78 -0.46
-2.75 0.88 -0.63
F
40 40 40 40
1818 2640 2990 3440
Ventral septal defect None None Ventral septal defect
53 54 54 50
-1.38 -2.70 -3.75 -0.46
-0.67 -2.58 -2.30 -0.12
-2.74 -6.43 -3.75 -0.08
F
Unknown
Unknown
None
Table 3. Current Status of Children with FAS and Autism as Compared with Children with Only FAS
Mental retardation Cerebral palsy Hyperactivity Mental retardation Mental retardation Mental retardation Hyperactivity Mental retardation Mental retardation Mental retardation Mental retardation Spastic cerebral palsy Mental retardation Seizures
35
30
46
1.64
1.20
-4.00
children may have suffered greater prenatal insult than did the non-autistic children. Estimates of maternal alcoFAS/ hol intake during pregnancy were not available so that it Autistic FAS Statistic P was not possible to determine whether the autistic FAS 2 (6) 2 (8) Chi Square NS Adopted ( N ) 1 (6) 0 (8) Chi Square NS Number of foster children had been exposed to greater amounts of alcohol placements >3? in utero. Prenatal alcohol exposure has not previously 6 (6) 4 (8) Chi Square
