PEDIATRICS Vol. 107 No. 2 February 2001, pp. 411-412
COMMENTARY:
Is There an Epidemic of Autism?
The debate on the hypothesis of a secular
increase in rates of autism would benefit from a clear recognition of
the methodologic limitations of existing data. No psychiatric case
register study has ever allowed for estimating and monitoring the
incidence of autistic conditions over time.1
Cross-sectional surveys hugely differ in their case definition and case
identification methods that account for large variations in prevalence
estimates both over time and across areas, precluding a meaningful
analysis of time trends. That rates in recent surveys are substantially
higher than 30 years ago merely reflects the adoption of a much broader
concept of autism, a recognition of autism among normally intelligent
subjects, changes in diagnostic criteria, and an improved
identification of persons with autism attributable to better
services.1,2 The only epidemiologic study where case
definition and identification could be held constant failed to detect
an increase in rates of autism in successive birth cohorts from 1972 to
1985.3
Most of the claims about the `epidemic' of autism are therefore based
on referral statistics from various centers. The report of the
Department of Developmental Services from California has been, and
still is, widely quoted as evidence for an epidemic of
autism.4 The key data of this report (Table
1 and Fig
1) need a critical examination.
TABLE 1
Autism and Other PDDs Registered in California
(1987-1998)
View larger version (27K):
[in a new window]
Fig. 1.
Distribution of birth dates of regional center eligible persons with
autism (California).
First, the figures apply to numbers rather than rates and fail to account for changes in the size and composition of the underlying population. Between 1987 and 1999, the population of California rose from 27 777 158 to 33 145 121 persons (+19.3%) and that of the 0- to 14-year-olds (the age group where most of the increase was reported) increased from 6 009 165 to 7 557 886 children (+25.8%). Positive migration fluxes to California both from within the United States and from abroad might also be differentially related to the presence of an autistic child in a family. None of these basic epidemiologic concerns were taken up in the report.
Second, no attempt was made to control for changes in diagnostic concepts and definitions. The report refers to definitions in the Diagnostic and Statistical Manual of the Mental Disorders, IV (DSM-IV) but completely ignores that, in view of the wide age range of the sample and study period (1987-1998), multiple diagnostic systems were in fact used over the years producing heterogeneity in diagnoses in successive birth cohorts. In 1980, DSM-III introduced the terminology of pervasive developmental disorder (PDD) widening the previous concept of infantile autism. The change from DSM-III to DSM-III-R in 1987 was associated with an additional broadening of the concept of PDD at the expense of its specificity. In 1994, the diagnostic categories of Asperger syndrome, Rett syndrome and childhood disintegrative disorder were introduced for the first time in DSM-IV as subcategories of PDDs; moreover, the diagnostic boundaries of pervasive developmental disorders not otherwise specified (PDD-NOS) were inadvertently widely broadened because of editorial changes in the layout of the diagnostic criteria.5 Not surprisingly, these 4 categories account for the largest increase in the California report (Table 1, Other PDD).
Third, in California and elsewhere, autistic children are now diagnosed at a much earlier age. A decreasing mean age at diagnosis will necessarily result in an increased number of reported cases, even assuming a stable prevalence or incidence. Thus, age-specific rates among older children are required to test time trends.
Fourth, the only comparison made in the report with an epidemiologic estimate was inaccurate. The report stated that the number (N = 1685) of new persons (of all ages) entering their database in 1998 exceeds that expected from one annual birth cohort; however, this comparison is nonsensical because it is confounded by age differences. More meaningful comparisons tell a very different story. Using a conservative estimate of 22.125/10 000 for all types of PDDs deriving from a recent review1 and the January 1999 census estimates (http://www.census.gov) for California for the whole population (N = 33 145 121) and subjects aged 0 to 19 (N = 9 916 162), the expected numbers of subjects with a PDD in California in 1998 are 73 334 and 21 940 persons, respectively. Furthermore, as of July 2000, the total number of persons with autism registered to California Developmental Services was 13 054. Thus, as one would predict with routine administrative statistics, the California reports seriously underestimate the size of the problem (Table 1); and surely our calculations provide no basis for the claim of an epidemic based on the reports' figures.
Fifth, far from being specific to autism, upward trends were also reported for other disorders (such as cerebral palsy, epilepsy, or mental retardation) for which there is no epidemiologic evidence for a secular increase. This points towards a general methodologic and recording artifact at the source of the reported increases.
Yet, the most worrying aspect of the California report lies in the distortion of data graphically portrayed in Fig 1. The report states that Fig 1 derives from the "... 1991 population of persons (7915) with autism," therefore clearly indicating the cross-sectional nature of the sample. Instead of age, the report plots year of birth on the horizontal axis thereby artificially creating the impression of a prospective data collection. The note of caution in the text (`Data points in Fig 1 do not show how many persons enter the system in a given year, but how many already in the system were born in a given year') has been obviously overlooked by most commentators. This graphical display deliberately transforms what is an age effect into what seems a cohort effect, although the latter cannot be tested with such cross-sectional data. By analogy, any sample with a marked skewed age-distribution (for example, take a survey of Army personnel) could be misleadingly portrayed by replacing age by year of birth and giving the same impression of an upward trend over time than in Fig 1 (but nobody would interpret the transformed personnel Army data as indicative of rising numbers of militaries!). The point is that, in both examples, there was no passage of time allowing inferences to be made on trends over time.
The misuse of these data by some investigators6 is another tribute to their poor research methodology. To date, the epidemiologic evidence for a secular increase in the incidence of PDDs is both meager and negative.1 We simply lack good data to test hypotheses on secular changes in the incidence of autism. Because of specific methodologic limitations, the high prevalence rates reported in recent autism surveys cannot be used to derive conclusions on this issue.1,2 Prevalence data nevertheless point to the magnitude of the problem, which had clearly been underestimated in the past. But there is no need to raise false alarms on putative epidemics nor to practice poor science to draw the attention to the unmet needs of large numbers of seriously impaired children and adults. More complex monitoring systems than those currently in place are needed to address the issue of secular changes in the incidence of PDDs. Maintaining case definition and identification constant, focusing on children in the upper range of school age years, controlling for changes in the population (ie, differential migration, etc... ) and relying on adequate sample sizes are required for future epidemiologic efforts in this area.
MRC Child Psychiatry Unit/Institute of Psychiatry
Denmark Hill
De Crespigny Park
London SE5 8AF, United Kingdom
FOOTNOTES
Received for publication Nov 20, 2000; accepted Nov 20, 2000.
Address correspondence to Eric Fombonne, MD, FRCPsych, MRC Child Psychiatry Unit/Institute of Psychiatry, Denmark Hill, De Crespigny Park, London SE5 8AF, United Kingdom. E-mail: e.fombonne{at}iop.kcl.ac.uk
ABBREVIATIONS
DSM-IV, Diagnostic and Statistical Manual of Mental Disorders, IV; PDD, pervasive developmental disorder; PDD-NOS, pervasive developmental disorder-not otherwise specified.
REFERENCES
- Fombonne E. The epidemiology of autism and related pervasive developmental disorders. In: Lord C, ed. Educating Children With Autism. Washington, DC: National Academy of Sciences Press; 2001. In press
- Fombonne E Epidemiological surveys of autism: a review. Psychol Med 1999; 29:769-786 [CrossRef][Medline]
- Fombonne E, du Mazaubrun C, Cans C, Grandjean H Autism and associated medical disorders in a large French epidemiological sample. J Am Acad Child Adolesc Psychiatry 1997; 36:1561-1569 [CrossRef][Medline]
- Department of Developmental Services. Changes in the Population of Persons With Autism and Pervasive Developmental Disorders in California's Developmental Services System: 1987 Through 1998. Report to the Legislature, March 1, 1999. Available at: http://www.dds.ca.gov
- Volkmar F, Shaffer D, First M PDD-NOS in DSM IV. J Autism Dev Disord 2000; 30:74-75 [Medline]
- Wakefield A MMR vaccination and autism. Lancet 1999; 354:949-950 [Medline]
Pediatrics (ISSN 0031 4005). Copyright ©2001 by the American Academy of Pediatrics
CiteULike 
Connotea 
Del.icio.us 
Digg 
Facebook 
Reddit 
Technorati 
Twitter What's this?
This article has been cited by other articles:
|
|
 |
|
  M. King and P. Bearman Diagnostic change and the increased prevalence of autism Int. J. Epidemiol., October 1, 2009; 38(5): 1224 - 1234. [Abstract] [Full Text] [PDF]
|
|
|
|
 |
|
 A.H.A. Latif and W.R. Williams Diagnostic trends in autistic spectrum disorders in the South Wales valleys Autism, November 1, 2007; 11(6): 479 - 487. [Abstract] [PDF]
|
|
|
|
 |
|
 D. L. Leslie and A. Martin Health Care Expenditures Associated With Autism Spectrum Disorders Arch Pediatr Adolesc Med, April 1, 2007; 161(4): 350 - 355. [Abstract] [Full Text] [PDF]
|
|
|
|
|
|
C. Silverman and J. P. Brosco Understanding Autism: Parents and Pediatricians in Historical Perspective Arch Pediatr Adolesc Med, April 1, 2007; 161(4): 392 - 398. [Abstract] [Full Text] [PDF]
|
|
|
|
|
|
A. E. Brachlow, K. K. Ness, M. L. McPheeters, and J. G. Gurney Comparison of Indicators for a Primary Care Medical Home Between Children With Autism or Asthma and Other Special Health Care Needs: National Survey of Children's Health Arch Pediatr Adolesc Med, April 1, 2007; 161(4): 399 - 405. [Abstract] [Full Text] [PDF]
|
|
|
|
|
|
H. O. Atladottir, E. T. Parner, D. Schendel, S. Dalsgaard, P. H. Thomsen, and P. Thorsen Time Trends in Reported Diagnoses of Childhood Neuropsychiatric Disorders: A Danish Cohort Study Arch Pediatr Adolesc Med, February 1, 2007; 161(2): 193 - 198. [Abstract] [Full Text] [PDF]
|
|
|
|
 |
|
 A. Hillier, T. Fish, P. Cloppert, and D. Q. Beversdorf Outcomes of a Social and Vocational Skills Support Group for Adolescents and Young Adults on the Autism Spectrum Focus on Autism and Other Developmental Disabilities, January 1, 2007; 22(2): 107 - 115. [Abstract] [PDF]
|
|
|
|
 |
|
 E. Fombonne, R. Zakarian, A. Bennett, L. Meng, and D. McLean-Heywood Pervasive Developmental Disorders in Montreal, Quebec, Canada: Prevalence and Links With Immunizations Pediatrics, July 1, 2006; 118(1): e139 - e150. [Abstract] [Full Text] [PDF]
|
|
|
|
 |
|
 J Leask, A Leask, and N Silove Evidence for autism in folklore? Arch. Dis. Child., March 1, 2005; 90(3): 271 - 271. [Full Text] [PDF]
|
|
|
|
 |
|
 S. Naqvi Review of Child and Adolescent Psychiatry Focus, October 1, 2004; 2(4): 529 - 540. [Abstract] [Full Text] [PDF]
|
|
|
|
|
|
H. C. Meissner, P. M. Strebel, and W. A. Orenstein Measles Vaccines and the Potential for Worldwide Eradication of Measles Pediatrics, October 1, 2004; 114(4): 1065 - 1069. [Abstract] [Full Text] [PDF]
|
|
|
|
|
|
S. K. Parker, B. Schwartz, J. Todd, and L. K. Pickering Thimerosal-Containing Vaccines and Autistic Spectrum Disorder: A Critical Review of Published Original Data Pediatrics, September 1, 2004; 114(3): 793 - 804. [Abstract] [Full Text] [PDF]
|
|
|
|
 |
|
 P. Ip, V. Wong, M. Ho, J. Lee, and W. Wong Mercury Exposure in Children With Autistic Spectrum Disorder: Case-Control Study J Child Neurol, June 1, 2004; 19(6): 431 - 434. [Abstract] [PDF]
|
|
|
|
|
|
F. DeStefano, T. K. Bhasin, W. W. Thompson, M. Yeargin-Allsopp, and C. Boyle Age at First Measles-Mumps-Rubella Vaccination in Children With Autism and School-Matched Control Subjects: A Population-Based Study in Metropolitan Atlanta Pediatrics, February 1, 2004; 113(2): 259 - 266. [Abstract] [Full Text] [PDF]
|
|
|
|
 |
|
 G. Baird, H. Cass, and V. Slonims Diagnosis of autism BMJ, August 30, 2003; 327(7413): 488 - 493. [Full Text] [PDF]
|
|
|
|
|
|
R Lingam, A Simmons, N Andrews, E Miller, J Stowe, and B Taylor Prevalence of autism and parentally reported triggers in a north east London population Arch. Dis. Child., August 1, 2003; 88(8): 666 - 670. [Abstract] [Full Text] [PDF]
|
|
|
|
|
|
J. G. Gurney, M. S. Fritz, K. K. Ness, P. Sievers, C. J. Newschaffer, and E. G. Shapiro Analysis of Prevalence Trends of Autism Spectrum Disorder in Minnesota Arch Pediatr Adolesc Med, July 1, 2003; 157(7): 622 - 627. [Abstract] [Full Text] [PDF]
|
|
|
|
|
|
C Cans, P Guillem, J Fauconnier, P Rambaud, and P S Jouk Disabilities and trends over time in a French county, 1980-91 Arch. Dis. Child., February 1, 2003; 88(2): 114 - 117. [Abstract] [Full Text] [PDF]
|
|
|
|
 |
|
 M. Yeargin-Allsopp, C. Rice, T. Karapurkar, N. Doernberg, C. Boyle, and C. Murphy Prevalence of Autism in a US Metropolitan Area JAMA, January 1, 2003; 289(1): 49 - 55. [Abstract] [Full Text] [PDF]
|
|
|
|
|
|
E. Fombonne The Prevalence of Autism JAMA, January 1, 2003; 289(1): 87 - 89. [Full Text] [PDF]
|
|
|
|
|
|
M. F Blaxill Any changes in prevalence of autism must be determined BMJ, February 2, 2002; 324(7332): 296 - 296. [Full Text]
|
|
|
|
|
|
L. Smeeth, A. J Hall, L. C Rodrigues, X. Huang, P. G Smith, E. Fombonne, M. Edwardes, M. Baltzan, F E. Yazbak, J. A Kaye, et al. Measles, mumps, and rubella (MMR) vaccine and autism BMJ, July 21, 2001; 323(7305): 163 - 163. [Full Text] [PDF]
|
|
|
|
|
|
S. Chakrabarti and E. Fombonne Pervasive Developmental Disorders in Preschool Children JAMA, June 27, 2001; 285(24): 3093 - 3099. [Abstract] [Full Text] [PDF]
|
|
|
|
|
|
S. L. Hyman, P. M. Rodier, and P. Davidson Pervasive Developmental Disorders in Young Children JAMA, June 27, 2001; 285(24): 3141 - 3142. [Full Text] [PDF]
|
|
|
|
|
|
N. A. Halsey, S. L. Hyman, and the Conference Writing Panel Measles-Mumps-Rubella Vaccine and Autistic Spectrum Disorder: Report From the New Challenges in Childhood Immunizations Conference Convened in Oak Brook, Illinois, June 12-13, 2000 Pediatrics, May 1, 2001; 107(5): 84e - 84. [Abstract] [Full Text]
|
|
|
|
|
|
L. Dales, S. J. Hammer, and N. J. Smith Time Trends in Autism and in MMR Immunization Coverage in California JAMA, March 7, 2001; 285(9): 1183 - 1185. [Abstract] [Full Text] [PDF]
|
|
| ||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
