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How Different Is Early-Onset Childhood Disintegrative Disorder from Autistic Disorder with Speech Loss?*

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How Different Is Early-Onset Childhood Disintegrative Disorder from Autistic Disorder with Speech Loss?* Open Journal of Psychiatry, 2013, 3, 39-45 OJPsych http://dx.doi.org/10.4236/ojpsych.2013.32A007 Published Online April 2013 (http://www.scirp.org/journal/ojpsych/) How different is early-onset childhood disintegrative * disorder from autistic disorder with speech loss? Hiroshi Kurita1,2#, Kanna Inoue3 1Department of Child Psychiatry, Zenkoku Ryoiku Sodan Center, Tokyo, Japan 2Department of Mental Health, Graduate School of Medicine, Tokyo University, Tokyo, Japan 3Department of Child Psychiatry, Nerima Welfare Center for Handicapped Persons, Tokyo, Japan Email: #[email protected] Received 27 February 2013; revised 30 March 2013; accepted 9 April 2013 Copyright © 2013 Hiroshi Kurita, Kanna Inoue. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. ABSTRACT Diagnosis; Regression To examine the difference between early-onset (< age 3) childhood disintegrative disorder (CDD) and au- 1. INTRODUCTION tistic disorder with speech loss (ADSL), 8 children Childhood disintegrative disorder (CDD) originates from with early-onset CDD (mean age = 7.6 years, SD = 3.8; Dementia infantilis first reported by Heller [1] in 1908 in 6 males) were compared with 92 age and gender-ratio six infants who had displayed profound mental regres- comparable children with ADSL (mean age = 6.8 sion during the third and fourth years of life. CDD is a years, SD = 4.1; 70 males) on 24 variables not directly subtype of pervasive developmental disorders (PDDs) in related to the key features of CDD (regression after DSM-IV [2] and ICD-10 [3], using similar diagnostic normal development for at least the first 2 years after criteria. Its salient feature is a marked regression result- birth). Compared with the ADSL group, the early- ing in an autistic and intellectually disabled state after onset CDD group had a tendency to have a higher apparently normal development for at least the first 2 rate of a psychosocial event before speech loss (SL) years after birth. Although studies on CDD are scarce (early-onset CDD, 75.0% vs ADSL, 37.0%, p = 0.057; due to its rarity (i.e. an estimated prevalence of 2.0/ effect size (phi) = 0.211, p < 0.05); a significantly 100,000 [4]), CDD is validated to a certain extent for its higher rate of fearfulness during SL (62.5% vs 4.3%, less favorable outcomes in childhood [5], adolescence, p = 0.000; phi = 0.551, p < 0.05); and a tendency to and adulthood [6,7], and in its higher incidence of epi- have a higher rate of epilepsy (25.0% vs 3.3%, p = lepsy in childhood [8], compared with autistic disorder 0.050; phi = 0.271, p < 0.05), a tendency to have a lower (AD). CDD is also distinct in its higher incidence of epi- rate of the Childhood Autism Rating Scale-Tokyo lepsy, more pronounced stereotypical behavior, and a Version (CARS-TV) total score ≥ 30 (75.0% vs 95.7%, less uneven intellectual profile in childhood compared p = 0.072; phi = 0.236, p < 0.05), and a significantly with AD with speech loss (SL) (ADSL), the type of AD lower rate of CARS-TV item 2 (imitation) score ≥ 2 that is closest to CDD [9]. (50.0% vs 82.6%, p = 0.049; phi = 0.221, p < 0.05) on DSM-5 [10] proposes to subsume CDD under the sin- the first visit. The two groups did not exhibit any sig- gle category of autism spectrum disorder (ASD) together nificant difference in the other 19 variables. The with three other DSM-IV PDDs (AD, Asperger’s disor- findings of no significant difference in the great ma- der and pervasive developmental disorder not otherwise jority and a significant difference in the small minor- specified (PDD-NOS)), with Rett’s disorder (RD) ex- ity of the 24 variables between the two groups support cluded. However, it is important to further characterize integrating CDD into regressive autism spectrum CDD, since many issues still remain unclear. One of disorder and studying CDD as its prototypical form. them is the difference between early-onset (< age 3) CDD and AD with regression. Twenty-two of 57 (38.6%) Keywords: Autistic Disorder; Autism Spectrum CDD cases in 14 studies [5,6,8,11-21] published in the Disorder (ASD); Childhood Disintegrative Disorder; last 25 years are early-onset, while 20% to 40% of AD *The authors declare that they have no conflict of interest. cases display regression [22-26]. #Corresponding author. Early-onset CDD cases satisfy not only CDD criteria OPEN ACCESS 40 H. Kurita, K. Inoue / Open Journal of Psychiatry 3 (2013) 39-45 A (normal for at least the first 2 years after birth), B (loss Table 1. Diagnostic criteria for early-onset childhood disinter- of acquired skills in at least two of the five areas) and C grative disorder (CDD). (abnormalities in at least two of the three areas: social A. Apparently normal development for at least the first 2 interaction impairment, communication impairment, and years after birth. restricted/repetitive behavior/interests (RRBI)), but also B. Loss of acquired skills in at least two of the five areas AD criterion B (onset < age 3) by definition of early- (language, social skills, bladder/bowel control, play and onset (< age 3). If early-onset CDD cases satisfy CDD motor skills) before the age of 3. criterion C by showing abnormalities in all three areas C. Abnormalities in at least two of the three areas (social instead of two, they are quite likely to also satisfy AD interaction impairment, communication impairment and criterion A (six or more symptom items in three areas, restricted/repetitive behavior/interests). including at least two from social interaction impairment D. With Rett’s disorder, Asperger’s disorder and schizophrenia excluded, the diagnosis of early-onset CDD but not autistic disorder and one each from communication impairment and or pervasive developmental disorder not otherwise specified in a RRBI). In such cases, the conclusive CDD diagnosis re- case that satisfies criteria A to C. quires that CDD criterion D (i.e. not better accounted for by another specific PDD or by schizophrenia) be met. onset CDD (mean age 7.6 years, SD = 3.8; 6 males, 2 Excluding RD, Asperger’s disorder, PDD-NOS or schi- females) and 92 children with ADSL (mean age 6.8 zophrenia is not difficult in such cases. However, a diag- years, SD = 4.1; 70 males, 22 females) not significantly nosis of early-onset CDD by excluding AD may not be different in age (t(98) = 0.522, p = 0.603) and gender the ultimate diagnosis, since a diagnosis of AD is still ratio (p = 1.000) from the 8 children with early-onset possible in the event that AD criterion C (not better ac- CDD. They were collected in the period 1982-2011 as counted for by RD or CDD) is considered to be met. This consecutive referrals to two leading facilities for deve- impasse remains in the absence of a rule by which it is lopmental disorders in Tokyo (the authors’ affiliations). determined that the diagnosis of CDD or AD takes prior- In each facility, a clinical team of experienced clinicians ity in such cases. In addition, another rule is needed to (i.e. child psychiatrists, pediatric neurologists, psycho- diagnose early-onset CDD but not PDD-NOS in cases logists and speech pathologists) lead by a child psychia- that satisfy the CDD criteria A to C and AD criterion B, trist (the first author) diagnosed children according to the but does not meet the AD criterion A. However, DSM- relevant ICD [3,27] and DSM criteria [2,28]. This retro- IV provides no such rules. Since we were aware of this spective chart review study, which was based on clinical shortcoming in CDD diagnosis in DSM-IV, we devised data accumulated on patients in our clinics over the last diagnostic criteria for early-onset CDD based on DSM- 30 years, was one part of a comprehensive study of the IV CDD criteria as shown in Table 1 soon after the pub- prognoses of PDDs approved by the ethics committee of lication of DSM-IV to keep early-onset CDD as an inte- the Tokyo University Graduate School of Medicine. gral part of CDD for CDD studies. In this study, an effort was made to clarify the differ- 2.2.1. Early-Onset CDD ence between early-onset CDD and regressive AD as re- Of the 8 children with early-onset CDD, 5 children who presented by ADSL, through a comparison of clinical visited either of the two facilities after the publication of variables not directly related to the key features of CDD DSM-IV were diagnosed by the clinical teams as having (regression after normal development for at least the first early-onset CDD according to the criteria in Table 1 by 2 years after birth). consensus at the time of their first visit. The remaining 3 children, who visited one of the two facilities before the 2. METHODS publication of DSM-IV, were diagnosed as having dis- 2.1. Diagnostic Criteria for Early-Onset CDD integrative psychosis based on ICD-9 [27]. The clinical teams reached a consensus from the details in the clini- Table 1 shows the diagnostic criteria for early-onset cal records and diagnosed these 3 children as having CDD. Criteria A to C are basically the same as those in early-onset CDD according to the criteria in Table 1. Ex- DSM-IV CDD criteria, except for adding “before the age perienced pediatric neurologists ruled out a neurological of 3” in criterion B to indicate the upper limit age at disease in the 8 children.
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