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The multiple faces of catatonia in autism spectrum disorders: descriptive clinical experience of 22 patients over 12 years

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Abstract

A retrospective review was conducted from the inpatient and outpatient records of twenty-two autistic youth presenting to a neurobehavioral service over a twelve-year period for combined psychiatric and behavioral pathology who also met DSM5 criteria for catatonia. Six autistic girls and 16 autistic boys ranging from ages eight to 26 years old were identified, and their variegated symptoms evaluated. Stereotypy, posturing, negativism, mutism and stupor were the most common catatonic symptoms, each present in more than half of the study patients. One patient had abnormal vital signs indicative of malignant catatonia. Twenty patients had concomitant repetitive self-injurious behaviors that had led to significant tissue injury and were refractory to psychotropic and behavioral interventions. The sample was weighted towards patients with severe self-injurious behavior, which often was the reason for admission. The many “faces” of catatonia in autism spectrum disorders are seen in this sample, and the novel recognition of repetitive self-injury as an under-recognized motor symptom of catatonia is highlighted. The preliminary findings in this study open many important future vistas for ongoing research regarding catatonia in ASDs.

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References

  1. Kahlbaum K (1874) Die Katatonie oder das Spannungsirresein. Verlag August Hirshwald, Berlin

    Google Scholar 

  2. American Psychiatric Association (2014) Diagnostic and statistical manual of mental disorders: DSM 5, 5th edn. American Psychiatric Association, Washington, DC

    Google Scholar 

  3. Morrison J (1973) Catatonia: retarded and excited types. Arch Gen Psychiatry 28:39–41

    Article  CAS  PubMed  Google Scholar 

  4. Bush G, Fink M, Petrides G, Dowling F, Francis A (1996) Catatonia: I. Rating scale and standardized examination. Acta Psychiatr Scand 93:129–136

    Article  CAS  PubMed  Google Scholar 

  5. Bush G, Fink M, Petrides G, Dowling F, Francis A (1996) Catatonia. II. Treatment with lorazepam and electroconvulsive therapy. Acta Psychiatr Scand 93:137–143

    Article  CAS  PubMed  Google Scholar 

  6. Fink M, Taylor A (2006) Catatonia: a clinician’s guide to diagnosis and treatment. Cambridge University Press, Cambridge

    Google Scholar 

  7. Caroff S, Mann S, Francis A, Fricchione G (2004) Catatonia: from psychopathology to neurobiology. American Psychiatric Publishing Inc, Washington

    Google Scholar 

  8. Ghaziuddin N, Marcotte K, Dhossche D (2012) Retrospective chart review of catatonia in child and adolescent psychiatric patients. Acta Psychiatr Scand 125(1):33–38

    Article  CAS  PubMed  Google Scholar 

  9. Wing L, Shah A (2000) Catatonia in autistic spectrum disorders. Br J Psychiatry 176:357–362

    Article  CAS  PubMed  Google Scholar 

  10. Billstedt E, Gilberg C, Gilberg C (2005) Autism after adolescence: population-based 13- to 22-year follow-up study of 120 individuals with autism diagnosed in childhood. J Autism Dev Disord 35:351–360

    Article  PubMed  Google Scholar 

  11. Breen J, Hare D (2017) The nature and prevalence of catatonic symptoms in young people with autism. J Intellect Disabil Res 61(6):580–593

    Article  CAS  PubMed  Google Scholar 

  12. Dhossche D, Wing L, Ohta M, Neumarker K-J (eds) (2006) Catatonia in Autism spectrum disorders, vol 72. International Review of Neurobiology. Elsevier Academic Press, San Diego, USA

    Google Scholar 

  13. Wachtel L, Contrucci-Kuhn S, Griffin M, Thompson A, Dhossche D, Reti I (2009) ECT for self-injury in an autistic boy. Eur Child Adolesc Psychiatry 18(7):458–463. https://doi.org/10.1007/s00787-009-0754-8

    Article  PubMed  Google Scholar 

  14. Wachtel L, Dhossche D (2010) Self-injury in autism as an alternate sign of catatonia: implications for electroconvulsive therapy. Med Hypotheses 75(1):111–114. https://doi.org/10.1016/j.mehy.2010.02.001

    Article  PubMed  Google Scholar 

  15. Wachtel L, Jaffe R, Kellner C (2011) Electroconvulsive therapy for psychotropic-refractory bipolar affective disorder and severe self-injury and aggression in an 11-year-old autistic boy. Eur Child Adolesc Psychiatry 20(3):147–152. https://doi.org/10.1007/s00787-010-0155-z

    Article  PubMed  Google Scholar 

  16. Wachtel L, Kahng S, Dhossche D, Cascella N, Reti I (2008) Electroconvulsive therapy for catatonia in an autistic girl. Am J Psychiatry 165:329–333

    Article  PubMed  Google Scholar 

  17. Wachtel L, Shorter E (2013) Self-injurious behaviour in children: a treatable catatonic syndrome. Aust N Z J Psychiatry 47(12):1113–1115. https://doi.org/10.1177/0004867413506500

    Article  PubMed  Google Scholar 

  18. Paclawskyj TR, Kurtz PF, O’Connor JT (2004) Functional assessment of problem behaviors in adults with mental retardation. Behav Modif 28(5):649–667

    Article  PubMed  Google Scholar 

  19. Soke GN, Rosenberg SA, Hamman RF, Fingerlin T, Robinson C, Carpenter L, Giarelli E, Lee LC, Wiggins LD, Durkin MS, DiGuiseppi C (2016) Brief report: prevalence of self-injurious behaviors among children with autism spectrum disorder-a population-based study. J Autism Dev Disord 46(11):3607–3614. https://doi.org/10.1007/s10803-016-2879-1

    Article  PubMed  PubMed Central  Google Scholar 

  20. Folch A, Cortes MJ, Salvador-Carulla L, Vicens P, Irazabal M, Munoz S, Rovira L, Orejuela C, Haro JM, Vilella E, Martinez-Leal R (2018) Risk factors and topographies for self-injurious behaviour in a sample of adults with intellectual developmental disorders. J Intellect Disabil Res. https://doi.org/10.1111/jir.12487

    Article  PubMed  Google Scholar 

  21. Wachtel L, Dhossche D (2010) Self-injury in autism as an alternative sign of catatonia. Med Hypotheses 75(1):111–114

    Article  PubMed  Google Scholar 

  22. Shorter E, Fink M (2018) The madness of fear: a history of catatonia. Oxford University Press, New York

    Book  Google Scholar 

  23. Wachtel LE, Kahng S, Dhossche DM, Cascella N, Reti IM (2008) ECT for catatonia in an autistic girl. Am J Psychiatry 165(3):329–333. https://doi.org/10.1176/appi.ajp.2007.07081246

    Article  PubMed  Google Scholar 

  24. Zaw F, Bates G, Murali V, Bentham P (1999) Catatonia, autism, and ECT. Dev Med Child Neurol 41:843–845

    Article  CAS  PubMed  Google Scholar 

  25. Ohta M, Kano Y, Nagai Y (2006) Catatonia in individuals with autism spectrum disorders in adolescence and early adulthood: a long-term prospective study. Int Rev Neurobiol 72:41–54

    Article  PubMed  Google Scholar 

  26. Hare D, Malone C (2004) Catatonia and autism spectrum disorders. Autism 8:183–195

    Article  PubMed  Google Scholar 

  27. Dhossche D (1998) Brief report: catatonia in autistic disorders. J Autism Dev Disord 28(4):329–331

    Article  CAS  PubMed  Google Scholar 

  28. Kakooza-Mwesige A, Wachtel L, Dhossche D (2008) Catatonia in autism: implications across the life span. Eur Child Adolesc Psychiatry 17(6):327–335

    Article  PubMed  Google Scholar 

  29. Wachtel L, Commins E, Park M, Rolider N, Stephens R, Reti I (2015) Neuroleptic malignant syndrome and delirious mania as malignant catatonia in autism: prompt relief with electroconvulsive therapy. Acta Psychiatr Scand 132(4):319–320. https://doi.org/10.1111/acps.12441

    Article  CAS  PubMed  Google Scholar 

  30. Wachtel L, Griffin M, Dhossche D, Reti I (2010) Brief report: electroconvulsive therapy for malignant catatonia in an autistic adolescent. Autism 14(4):349–358. https://doi.org/10.1177/1362361309350135

    Article  PubMed  Google Scholar 

  31. Wachtel L, Griffin M, Reti I (2009) Electroconvulsive therapy in a man with autism experiencing severe depression, catatonia and self-injury. J ECT 26(1):70–73

    Article  Google Scholar 

  32. Wachtel L, Reti I, Ying H (2013) Stability of intraocular pressure after retinal reattachment surgery during electroconvulsive therapy for intractable self-injury in a 12-year-old autistic boy. J ECT 30(1):73–76

    Article  CAS  Google Scholar 

  33. Wachtel L, Schuldt S, Ghaziuddin N, Shorter E (2013) The potential role of electroconvulsive therapy in the ‘Iron Triangle’ of pediatric catatonia, autism, and psychosis. Acta Psychiatr Scand 128(5):408–409

    Article  CAS  PubMed  Google Scholar 

  34. Dhossche D, Shah A, Wing L (2006) Blueprints for the assessment, treatment, and future study of catatonia in autism spectrum disorders. Int Rev Neurobiol 72:267–284

    Article  PubMed  Google Scholar 

  35. Fink M, Taylor M, Ghaziuddin N (2006) Catatonia in autistic spectrum disorders: a medical treatment algorithm. Int Rev Neurobiol 72:233–244

    Article  PubMed  Google Scholar 

  36. Dhossche D (2004) Autism as early expression of catatonia. Med Sci Monit 10:RA31–RA39

    PubMed  Google Scholar 

  37. Dhossche D, Rout U (2006) Are autistic and catatonic regression related? A few working hypotheses involving GABA, Purkinje cell survival, neurogenesis, and ECT. Int Rev Neurobiol 72:55–79

    Article  PubMed  Google Scholar 

  38. Dhossche D, Song Y, Liu Y-M (2005) Is there a connection between autism, Prader-Willi syndrome, catatonia, and GABA? Int Rev Neurobiol 71:189–216

    Article  CAS  PubMed  Google Scholar 

  39. Dhossche DM, Bouman NH (1997) Catatonia in an adolescent with Prader-Willi syndrome. Ann Clin Psychiatry 9(4):247–253

    Article  CAS  PubMed  Google Scholar 

  40. Faedda G, Wachtel L, Higgins AM, Shprintzen R (2015) Catatonia in an adolescent with velo-cardio-facial syndrome. Am J Med Genet A 167A(9):2150–2153

    Article  PubMed  Google Scholar 

  41. Winarni T, Schneider A, Ghaziuddin N, Seritan A, Hagerman R (2015) Psychosis and catatonia in fragile X: case report and literature review. Intractable Rare Dis Res 4(5):139–146

    Article  PubMed  PubMed Central  Google Scholar 

  42. Shorter E, Wachtel L (2013) Childhood catatonia, autism and psychosis past and present: is there an ‘iron triangle’? Acta Psychiatr Scand 128(1):21–33. https://doi.org/10.1111/acps.12082

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  43. Stauder K (1934) Die todliche Katatonie. Arch Psychiatr Nervenkrank 102:614–634

    Article  Google Scholar 

  44. Dhossche D, Shettar S, Kumar T, Burt L (2009) Electroconvulsive therapy for malignant catatonia in adolescence. South Med J 102(11):1170–1172

    Article  PubMed  Google Scholar 

  45. Wachtel L, Crawford T, Dhossche D, Reti I (2010) Electroconvulsive therapy for pediatric malignant catatonia with cerebellar dysgenesis. Pediatr Neurol 43(6):427–430. https://doi.org/10.1016/j.pediatrneurol.2010.06.009

    Article  PubMed  Google Scholar 

  46. Ghaziuddin N, Hendriks M, Patel P, Wachtel LE, Dhossche DM (2017) Neuroleptic malignant syndrome/malignant catatonia in child psychiatry: literature review and a case series. J Child Adolesc Psychopharmacol 27(4):359–365. https://doi.org/10.1089/cap.2016.0180

    Article  PubMed  Google Scholar 

  47. Handen BL, Mazefsky CA, Gabriels RL, Pedersen KA, Wallace M, Siegel M, Autism, Developmental Disorders Inpatient Research C (2018) Risk factors for self-injurious behavior in an inpatient psychiatric sample of children with autism spectrum disorder: a naturalistic observation study. J Autism Dev Disord. https://doi.org/10.1007/s10803-017-3460-2

    Article  PubMed  PubMed Central  Google Scholar 

  48. Kahng S, Iwata B, Lewin A (2002) Behavioral treatment of self-injury, 1964–2000. Am J Ment Retard 107(3):212–221

    Article  PubMed  Google Scholar 

  49. Hanley G, Iwata B, McCord B (2003) Functional analysis of problem behavior: a review. J Appl Behav Anal 36(2):147–185

    Article  PubMed  PubMed Central  Google Scholar 

  50. Mace F, Mauk J (1995) Bio-behavioral diagnosis and treatment of self-injury. Ment Retard Dev Disabil 1:104–110

    Article  Google Scholar 

  51. Wachtel L, Hagopian L (2006) Psychopharmacology and applied behavioral analysis: tandem treatment of severe problem behaviors in intellectual disability and a case series. Isr J Psychiatry Relat Sci 43(4):265–274

    PubMed  Google Scholar 

  52. Bates W, Smeltzer D (1982) Electroconvulsive treatment of psychotic self-injurious behavior in a patient with severe mental retardation. Am J Psychiatry 139:1355–1356

    Article  CAS  PubMed  Google Scholar 

  53. Fink M (1999) Electroshock: healing mental illness. Oxford University Press, London

    Google Scholar 

  54. Carr V, Dorrington C, Schrader G, Wale J (1983) The use of ECT for mania in childhood bipolar disorder. Br J Psychiatry 143:411–415

    Article  CAS  PubMed  Google Scholar 

  55. Black D, Wilcox J, Stewart M (1985) The use of ECT in children: case-report. J Clin Psychiatry 46:98–99

    CAS  PubMed  Google Scholar 

  56. Cizadlo B, Wheaton A (1995) Case study: ECT treatment of a young girl with catatonia. J Am Acad Child Adolesc Psychiatry 34:332–335

    Article  CAS  PubMed  Google Scholar 

  57. Chung A, Varghese J (2008) Treatment of catatonia with electroconvulsive therapy in an 11 year-old girl. Aust NZJ Psychiatry 42:251–253

    Article  Google Scholar 

  58. Arora M, Praharaj S, Prakash R (2008) Electroconvulsive therapy for multiple major self-mutilations in bipolar psychotic depression. Turk J Psychiatry 19(2):1–4

    Google Scholar 

  59. Wachtel LE, Shorter E, Fink M (2018) Electroconvulsive therapy for self-injurious behaviour in autism spectrum disorders: recognizing catatonia is key. Curr Opin Psychiatry 31(2):116–122. https://doi.org/10.1097/YCO.0000000000000393

    Article  PubMed  Google Scholar 

  60. DeJong H, Bunton P, Hare DJ (2014) A systematic review of interventions used to treat catatonic symptoms in people with autistic spectrum disorders. J Autism Dev Disord 44(9):2127–2136. https://doi.org/10.1007/s10803-014-2085-y

    Article  PubMed  Google Scholar 

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Acknowledgements

The author would like to thank Max Fink, MD and Edward Shorter, PhD for their invaluable and extensive mentorship, and the patients and their families who have so generously shared their stories.

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Correspondence to Lee Elizabeth Wachtel.

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Wachtel, L.E. The multiple faces of catatonia in autism spectrum disorders: descriptive clinical experience of 22 patients over 12 years. Eur Child Adolesc Psychiatry 28, 471–480 (2019). https://doi.org/10.1007/s00787-018-1210-4

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