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ORIGINAL PAPER

Asperger Syndrome and Autism: A Comparative LongitudinalFollow-Up Study More than 5 Years after Original Diagnosis

Mats Cederlund

Bibbi Hagberg

Eva Billstedt

I. Carina Gillberg

Christopher Gillberg

Published online: 6 March 2007

Springer Science+Business Media, LLC 2007

Abstract

Prospective follow-up study of 70 males withAsperger syndrome (AS), and 70 males with autismmore than 5 years after original diagnosis. Instrumentsused at follow-up included overall clinical assessment,the Diagnostic Interview for Social and Communica-tion Disorders, Wechsler Intelligence Scales, VinelandAdaptive Behavior Scales, and Global Assessment of Functioning Scale. Speciﬁc outcome criteria were used.Outcome in AS was good in 27% of cases. However,26% had a very restricted life, with no occupation/activity and no friends. Outcome in the autism groupwas signiﬁcantly worse. Males with AS had worse out-comes than expected given normal to high IQ. How-ever, outcome was considerably better than for thecomparison group of individuals with autism.

Keywords

Asperger syndrome

Autism

Follow-up

Intellectual ability

Outcome

DISCO

Introduction

Asperger syndrome (AS), described as ‘‘autistic psy-chopathy’’alreadyin1944bytheAustrianpaediatricianHans Asperger (Asperger,1944), did not appear asa diagnostic entity until the early 1980s after beingreintroduced by Lorna Wing (Wing,1981). The first setof diagnostic criteria were formulated by Gillberg andGillberg (1988/1989). The Diagnostic and StatisticalManual of Mental Disorders (DSM-IV), and the ICD-10 classification of Mental and Behavioural Disorders,did not publish diagnostic criteria for AS until well intothe 1990s (APA,1994, WHO,1993). These criteria have been widely criticized (e.g., Leekam, Libby,Wing, Gould, & Gillberg,2000; Miller & Ozonoff,1997), and there is still no consensus as to how ASshould best be delineated. The Gillberg´ s six criteria formaking the diagnosis of AS are based on HansAsperger´ s original publication and require majorproblems with social interaction (e.g., impairing diffi-culties making friends and understanding social cues),narrow interests, repetitive routines, speech and lan-guage (e.g., odd ways of using expressive language, oddspeech, intonation problems, and difficulties under-standing others), non-verbal communication problems,and motor clumsiness. At least 9 symptoms are re-quired for a diagnosis of AS. The DSM-IV criteria forAS specify impairments in social interaction (definedas for autism), restricted, repetitive, stereotypedbehaviour (defined as for autism), and absence of clinically significant delay in language or cognitivedevelopment, including self-help skills, adaptivebehaviour and curiosity about the environment, in thefirst 3 years of life (see Appendices). Only 3 symptomsare required for a diagnosis of AS.Over the years, there has been much discussionabout whether or not AS and so-called High Func-tioning Autism (HFA) are separate, similar or identi-cal conditions (Gillberg,1998; Mesibov, Kunce, &

M. Cederlund (

)

B. Hagberg

E. Billstedt

I. C. Gillberg

C. GillbergDepartment of Child and Adolescent Psychiatry, Institute of Neuroscience and Physiology, Go ¨ teborg University,Go ¨ teborgKungsgatan 12, 411 19, Swedene-mail: mats.cederlund@vgregion.seM. Cederlund

B. Hagberg

E. Billstedt

I. C. Gillberg

C. GillbergQueen Silvia´ s Child- and Adolescent Hospital,Otterha ¨ llegatan, 12 A, 411 18 Go ¨ teborg, Sweden

123

J Autism Dev Disord (2008) 38:72–85DOI 10.1007/s10803-007-0364-6

Schopler,1998; Wing & Potter, 2002). Although highverbal intelligence—including better verbal than per-formance skills on the Wechsler scales—and earlierlanguage development have been noted to be morecommon in people with AS than in classic autism, noresearch has so far been able to show clear-cut differ-ences between people diagnosed with HFA as com-pared with those who have been given a diagnosis of AS (Eisenmajeret al.,1998; Gilchrist et al.,2001; Howlin,2003).Very few reports on the outcome of AS have beenpublished. They have referred to small or highly se-lected clinical case samples without comparison groupsand have reported low levels of employment and socialfunctioning (Green, Gilchrist, Burton, & Cox,2000;Tantam,1991; Tsatsanis,2003; Wing,1981). In this study psychosocial functioning includes aspects of employment/studying, relationship to others, indepen-dent living, and absence of psychiatric problems.Intellectual decline over time, as measured by theWechsler scales, was reported in one study of theintermediate term outcome of AS (Nyde´ n, Billstedt,Hjelmqvist, & Gillberg,2001), but has not been ob-served in later studies. A tendency towards closing of the gap between superior verbal and inferior non-ver-bal skills over time has been observed (Szatmari, 2003,personal communication). Recent studies of the short-term outcome of AS have suggested a substantiallybetter outcome than in autism, which may have beendue to earlier and more effective interventions, or toother factors (Starr, Szatmari, Bryson, & Zweigenbaum,2003; Szatmari, Bryson, Boyle, Streiner, & Duku,2003; Tsatsanis,2003).Outcome in classic cases of autism has been inves-tigated in a number of studies in the past. The rate of poor or very poor psychosocial outcome (isolated lifewith high degree of dependency on others) has beenaround 50% (e.g., Gillberg & Steffenburg,1987;Howlin, Mawhood, & Rutter,2000; Howlin, Goode,Hutton, and Rutter,2004), and IQ has decreased overtime (Billstedt, Gillberg, & Gillberg,2005).The present study is probably the ﬁrst ever topresent a long-term perspective on the natural out-come of a reasonably large group of males with AS.The background and associated factors of the ASgroup have been outlined in two previous publications(Cederlund & Gillberg,2004; Gillberg & Cederlund,2005). The AS group was contrasted with a similarlyaged group of males with classic autism/atypical autismfollowed up using identical/similar instruments.One aim of the study was to examine the diagnosticstability over time within the autism spectrum, in thiscase the stability of AS as diagnosed on the basis of thecriteria formulated by Gillberg and Gillberg (1989) andGillberg (1991), and of autistic disorder and atypicalautism as diagnosed according to the DSM-IV. An-other aim was to estimate the frequency of severepsychiatric disorder (psychosis) in the investigatedgroups, given earlier reports of a high frequency of such disorders within the autism spectrum (e.g., Wozniaket al.,1997; Wolff,1995). Finally, there has been con- troversy about the rate of criminal offence within theAS/HFA group particularly after the publication of reports documenting a high rate of these diagnosesamong highly selected samples of offending psychiatricpatients (e.g., Siponmaa, Kristiansson, Jonsson, Nyde´ n,& Gillberg,2001; Scragg & Shah,1994), and so our third aim was to examine the prevalence of such of-fence committed by individuals with AS and autism inearly adult life.Some individuals with a diagnosis of Asperger syn-drome in childhood appear to do well in adult life, andthis has led some people to question the appropriate-ness of making the diagnosis at all. Could the diagnosisof Asperger syndrome actually contribute to problems(such as the supposed stigma of having a diagnosis atall) in certain cases? We consider that, if diagnoses areonly made in individuals who have clinical impairment,this would not be an issue. In the present study we usedthe Global Assessment of Functioning (GAF) scale toscore psychosocial functioning, so as to determinewhether or not an individual, who fulﬁls

symptom

cri-teria for AS should also be given the

diagnosis

of AS.The following hypotheses were tested: (i) diagnoseswithin the autism spectrum are stable over time, (ii)AS has a psychosocially better outcome than autism,(iii) better outcome in AS is attributable to higher IQ,(iv) intellectual ability declines over time in these dis-orders, (v) individuals with higher verbal IQ have thebest psychosocial outcome, (vi) those with an earlierdiagnosis have fewer problems in early adult life, (vii)there is a higher frequency of severe psychiatric dis-orders (psychosis), than in the general population,(viii) involvement with the police and the law is at thesame rate as in the general population.

Methods

AS groupOne-hundred males with AS, 16–36 years old, diag-nosed at the Child Neuropsychiatric Clinic (CNC) inGo ¨ teborg, Sweden, in the years 1985–1999, were ap-proached for inclusion in the follow-up study. Theywere born 1967–1988, and had been diagnosed with AS

J Autism Dev Disord (2008) 38:72–85 73

123

at ages 5.5–24.5 years. Mean age at diagnosis was11.3 years (

3.8 years), which is in keeping withprevious ﬁndings (Howlin & Ashgarian,1999). They allfulﬁlled the Gillberg and Gillberg criteria for AS(Gillberg1991; Gillberg & Gillberg,1989), which had been in use at the clinic since 1985.Criteria for inclusion in the follow-up study were (i)normal intelligence (IQ > 70) at diagnosis on theWechsler scales (WISC-R (n = 40), WISC-III (n = 52)or WAIS-R (n = 8)), (ii) AS diagnosis made

‡

5 yearsprior to follow-up, and (iii) age

‡

16 years at the censusdate 30/06/2004. We included males only, since nomore than 7 females meeting inclusion criteria werefound. The AS group had been assessed on theWechsler scales (Wechsler,1974,1992a,1992b) at original diagnosis, and had had the following meanresults: Full Scale Intelligence Quotient (FSIQ) 101.4(

18.3), Verbal Intelligence Quotient (VIQ) 107.2(

18.6) and Performance Intelligence Quotient(PIQ) 94.6 (

18.7) (VIQ > PIQ,

< 0.01).Sixty-one individuals in the AS group had had nofurther contact with the CNC after the diagnosis hadbeen made and information about results had beenshared with the family. A further 8 had had no contactwith the CNC for the past 5 years, but 31 had had oneor several contacts in the recent past (i.e., 2001 orlater). We have argued elsewhere (Cederlund & Gillberg,2004) that the 100 individuals approached are fairlyrepresentative of all males with AS, whose parentsapplied for clinical assessment and help for them in thelate 1980s and throughout the 1990s.Autism GroupA group of 16 to 38-year-old males from a group withautistic disorder (AD) diagnosed with autism (n = 62)or atypical autism (n = 22, including 2 with disinte-grative disorder) using the DSM-III (APA, 1980), andDSM-III-R (APA, 1987) criteria, before 10 years of age and followed-up with a similar protocol as the ASgroup at the CNC 1999–2002 (Billstedt, Gillberg, &Gillberg,2005; Billstedt, 2006, personal communica-tion), were used as a contrast group. These 84 indi-viduals were all the males from a community-basedstudy of autism, who were approached for inclusion ina longitudinal prospective follow-up study designedand carried out by the last three authors. We considerthis autism group to be representative of all individualswith autism diagnosed in the 1960s, 1970s and 1980s inthe community. The group was strikingly differentfrom the AS group in terms of IQ, but they still rep-resented a reasonable contrast group vis-a `-vis our ASgroup as regards their social impairment. The Autismgroup had been evaluated in childhood with the fol-lowing results: Severe Mental Retardation (SMR,IQ < 50) (n = 39), Mild Mental Retardation (MMR,IQ 50–69) (n = 31), Near Average Intelligence (NA,IQ 70–84) (n = 10) and Average Intelligence(A, IQ 85–114) (n = 4).Attrition

AS Group

There was no mortality in the original AS group of 100males. A letter of information about the AS study wassent to all 100 individuals or their parents (in thoseunder 18 years of age). The study was referred to as ‘‘afollow-up study of Asperger syndrome’’. Of the 100males with AS and/or their parents targeted for inclu-sion, 24 refused or failed all participation in the follow-up study. Nineteen of these declined participation overthe telephone, and three did so in writing. In two casesthere was no response in spite of reminders, and so wehave no clue as to who was ‘‘responsible’’ for thefailure to participate. (Of the 24 who refused, five hadnever been informed about their condition. Parentshad never told their child about the diagnosis, and theydid not want for their sons to find out about it now. Inthe other 19 cases, we only have limited informationabout the reason for refusal/failure to participate).There was no significant difference in mean FSIQ atoriginal diagnosis between the participating (n = 76),and the non-participating (n = 24) AS groups.

Autism Group

In the original autism group of 84 males, three (4%)had died before follow-up. The causes of death were:death in an accidental ﬁre (boy with Fragile X syn-drome and autism at 11 years of age); death in com-plications after heart surgery (boy with trisomy 13 anda severe heart malformation and autism, age at deathunknown); unknown date and cause of death (boy withatypical autism, age at death unknown). An additional4 declined participation, leaving 77 for possible inclu-sion in the Autism study group. Of those who declinedparticipation two had MMR and two SMR.Participants

AS Study Group

Of the 76 cases participating in the follow-up study,seventy had had a complete DISCO interview per-formed, and these were included for further study,