Table 1.
Main characteristics of the included studies regarding catatonia and ASD.
| Study | Study type and design (NOS) | Type of sample | Country | Sample | Diagnostic criteria | Mean age (SD) | Gender %Males | Key findings |
|---|---|---|---|---|---|---|---|---|
| Billstedt et al. [37] | Longitudinal (6) | Population-based | Sweden | 108 ASD | DSM-III/DSM-IV/ICD-10/HBSS/CARS/ABC/Wing criteria/DISCO | 25.5 | 70 | ASD subjects showed a poor outcome at follow-up. Childhood IQ-level was positively correlated with better adult outcome, as was the existence of some communicative phrase speech at age 6 years. Those diagnosed with catatonia had severe motor initiation problems |
| 13 Catatonia | ||||||||
| Breen and Hare [38] | Cross-sectional (5) | Population-based | UK | 99 ASD | ABQ | 15.7 | 79.8 | Attenuated behavior indicative of catatonia was common in young people with ASD. Six catatonic core symptoms were commonly reported, with difficulty initiating movement being least reported and physical and/or verbal prompts required most reported |
| 20 Catatonia | ||||||||
| Ghaziuddin et al. [33] | Cross-sectional (7) | Clinic-based | US | 81 ASD | Clinical criteria | 13.9 (2.04) | 80.2 | Subjects with catatonia were older and had a lower score of global functioning. Aggression was more common among the group diagnosed with catatonia compared with the noncatatonic group. Similarly, reduced movements and loss of speech were also more common among this group |
| 18 Catatonia | ||||||||
| Hare et al. [45] | Cross-sectional (5) | Population-based | UK | 99 ASD | ABQ | 15.7 | 79.8 | Catatonia appears to be more prevalent in both ASD and in genetic syndromes that are strongly associated with comorbid ASD than was previously thought |
| 20 Catatonia | ||||||||
| Hutton et al. [40] | Longitudinal (5) | Population-based | UK | 135 ASD | ADI-R/ADOS/SAPPA/ICD-10 | na | 77 | The presence of obsessive–compulsive behavior and of catatonia, which mainly seemed to stem from obsessive–compulsive symptoms, might be particularly characteristic of ASD. Of 135 individuals with ASD, 16% reported a new psychiatric disorder, of whom three patients reported a catatonia |
| 3 Catatonia | ||||||||
| Ohta et al. [44] | Longitudinal (4) | Clinic-based | Japan | 11 ASD | DSM-IV/ICD-10/Wing criteria | 27.6 (5.5) | 100 | The average age at onset of catatonia was 19 years (ranged 15–23). A total of 11.6% ASD had catatonia. Before the manifestation of a typical catatonic symptom, eight cases had prodromal symptoms, typically a gradually emerging lethargy with compulsive behaviors lasting for more than 1 year |
| 2 Catatonia | ||||||||
| Périsse et al. [43] | Cross-sectional (6) | Clinic-based | France | 29 ASD | ICD-10 /CARS | 14.8 (1.3) | 79.3 | Disruptive behaviors among adolescents with ASD may stem from diverse risk factors, including environmental problems, comorbid acute psychiatric conditions, or somatic diseases |
| 2 Catatonia | ||||||||
| Wachtel [34] | Longitudinal (4) | Clinic-based | US | 22 ASD | BFCRS | 8–25 | 72.72 | Multiple symptoms of catatonia are found in patients with ASD. Some of the most severe presentations include psychomotor excitement in the form of repetitive self-injury. The use of short-term and maintenance ECT might be helpful |
| 18 Catatonia | ||||||||
| Wachtel [36] | Longitudinal (4) | Clinic-based | US | 22 ASD | DSM-5 | 8–26 | 72.72 | The most common catatonic symptoms were agitation, stereotypy, posturing, negativism, mutism, stupor and grimacing. 91% ASD patients displayed repetitive self-injury |
| 22 Catatonia | ||||||||
| Wachtel [35] | Longitudinal (5) | Clinic-based | US | 22 ASD | DSM-5 | 8–26 | 72.72 | Benzodiazepines were of unclear benefit in the resolution of catatonia in ASD. Reasons for discontinuation included no benefit in terms of catatonic symptom reduction (31.8%), partial catatonic symptom reduction (40.9%), sedation (13.6%), and behavioral worsening (9.1%). A total of 76% patients who completed a trial of lorazepam, demonstrated catatonic symptom resolution. Four patients who failed lorazepam therapy responded to ECT. Maintenance electroconvulsive therapy was necessary for sustained symptom remission |
| 22 Catatonia | ||||||||
| Wing and Shah [41] | Cross-sectional (7) | Clinic-based | UK | 506 ASD | ICD-10/DISCO/Wing criteria | 24.9 | 82 | Catatonia onset ranged 10–19 years. Precipitating factors were suggested. From the onset of catatonia, 10% experienced a slow but steady deterioration in mobility and practical skills. Most of the abnormalities of movement resulted in slowing or stopping activities. There was an excess of males, more marked among those with catatonia. A total of 50% with catatonia-like deterioration were passive in social interaction. Catatonia was seen in a higher proportion of those with learning disabilities and language impairment |
| 30 Catatonia | ||||||||
| Wing and Shah [42] | Cross-sectional (7) | Clinic-based | UK | 200 ASD | DISCO/Wing criteria | 12.7 (8.1) | 83.5 | The most frequent catatonic symptoms reported from the DISCO were lack of facial expression, delayed echolalia, odd intonation, lack of cooperation, and poor eye contact. The lowest frequencies were found among those concerning fascination with visual stimuli. Higher IQ tended to be associated with less marked manifestations of the item concerned |
| 7 Catatonia | ||||||||
| 36 ASD | 2.8–11.6 | 72 | ASD was rated with more marked problems than the other three groups on DISCO. More children with learning disability had marked or moderate problems than those with specific language impairment, except for delayed echolalia, noisiness, and aggressiveness, which happened more often in those with specific language impairments. ASD individuals displayed more anxiety symptoms, more relevant in those with IQ > 70 | |||||
| 17 Learning disabled | ||||||||
| 14 Language impaired | ||||||||
| 15 HC | ||||||||
| 506 ASD | 24.9 | 82 | A total of 6% individuals with ASD had catatonia, 17% of those were > 15 years. Impairment of social interaction was one of the main outcomes. 50% with catatonia were passive in social interaction and were also more impaired in expressive language | |||||
| 30 Catatonia |
Abbreviations: ABC, the Autistic Behavior Checklist; ABQ, the Attenuated Behavior Questionnaire; ADI-R, the Autism Diagnostic Interview-Revised; ADOS, Autism Diagnostic Observation Schedule; ASD, Autism Spectrum Disorder; BFCRS, Bush–Francis Catatonia Rating Scale; CARS, the Childhood Autism Rating Scale; DISCO, the DIagnosis of Social and COmmunication Disorder Schedule; DSM-III, Diagnostic and Statistical Manual of Mental Disorders, third edition; DSM-IV, Diagnostic and Statistical Manual of Mental Disorders, fourth edition; DSM-5, Diagnostic and Statistical Manual of Mental Disorders, fifth edition; ECT, electroconvulsive therapy; HBSS, the MRC Handicaps, Behavior & Skills (HBS) schedule; HC, healthy control; ICD-10, International Classification of Diseases 10th Revision; NOS, Newcastle-Ottawa Scale for the evaluation of longitudinal and cross-sectional studies; SAPPA, the Schedule for Assessment of Psychiatric Problems Associated with Autism.