Catatonia in Down syndrome: systematic approach to diagnosis, treatment and outcome assessment based on a case series of seven patients

Judith H Miles,1,2 Nicole Takahashi,2 Julie Muckerman,2 Kerri P Nowell,2,3 Muaid Ithman4 1Department of Child Health, University of Missouri Healthcare, Columbia, MO, USA; 2Thompson Center for Autism and Neurodevelopmental Disorders, University of Missouri, Columbia, MO, USA; 3Department of Health P...

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Autores principales:	Miles JH, Takahashi N, Muckerman J, Nowell KP, Ithman M
Formato:	article
Lenguaje:	EN
Publicado:	Dove Medical Press 2019
Materias:	Lorazepam Electroconvulsive Therapy Dextromethorphan/quinidine Benzodiazepines Trisomy 21 Bush-Francis Catatonia Rating Scale. Neurosciences. Biological psychiatry. Neuropsychiatry RC321-571 Neurology. Diseases of the nervous system RC346-429
Acceso en línea:	https://doaj.org/article/eb1875e19a184d488522e2a0c856e362
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spelling	oai:doaj.org-article:eb1875e19a184d488522e2a0c856e3622021-12-02T04:09:53ZCatatonia in Down syndrome: systematic approach to diagnosis, treatment and outcome assessment based on a case series of seven patients1178-2021https://doaj.org/article/eb1875e19a184d488522e2a0c856e3622019-09-01T00:00:00Zhttps://www.dovepress.com/catatonia-in-down-syndrome-systematic-approach-to-diagnosis-treatment--peer-reviewed-article-NDThttps://doaj.org/toc/1178-2021Judith H Miles,1,2 Nicole Takahashi,2 Julie Muckerman,2 Kerri P Nowell,2,3 Muaid Ithman4 1Department of Child Health, University of Missouri Healthcare, Columbia, MO, USA; 2Thompson Center for Autism and Neurodevelopmental Disorders, University of Missouri, Columbia, MO, USA; 3Department of Health Psychology, University of Missouri Healthcare, Columbia, MO, USA; 4Department of Psychiatry, University of Missouri Health Care, Columbia, MO, USACorrespondence: Judith H MilesThompson Center for Autism and Neurodevelopmental Disorders, 205 Portland Street, Columbia, MO 65211, USATel +1 573 884 6838Fax +1 573 884 1151Email milesjh@missouri.eduObjective: The goal is to expand our knowledge of catatonia occurring in adolescents and young adults with Down syndrome (DS) by describing the first prospective, consecutive, well-characterized cohort of seven young people with DS diagnosed with catatonia and treated between 2013 and 2018, and to assess each patient’s treatment responses. Longitudinal assessment of each patient’s response to treatment is intended to provide clinicians and psychiatrists a firm foundation from which assess treatment efficacy.Study design: Young adults with Down syndrome were consecutively enrolled in the study as they were diagnosed with catatonia. A comprehensive data set included medical, laboratory, developmental, demographic, family, social and genetic data, including query into disorders for which individuals with DS are at risk. Catatonia was diagnosed based on an unequivocal history of regression, positive Bush-Francis Catatonia Rating Scale and positive response to intravenous lorazepam. Patients’ longitudinal progress was monitored using the Catatonia Impact Scale (CIS) developed for this purpose.Results: Seven consecutive DS patients, who presented with unequivocal regression were diagnosed with catatonia and treated for 2.7–6 years using standard-of-care therapies; primarily GABA agonist, lorazepam, electroconvulsive therapy (ECT) and glutamate antagonists (dextromethorphan/quinidine, memantine, minocycline). Responses to each treatment modality were assessed at clinic visits and through weekly electronic CIS reports.Conclusion: Seven young adults with DS were diagnosed with catatonia; all responded to Lorazepam and/or ECT therapy with good to very good results. Though ECT most dramatically returned patients to baseline, symptoms often returned requiring additional ECT. Dextromethorphan/quinidine, not used until mid-2017, appeared to reduce the reoccurrence of symptoms following ECT. Though all seven patients improved significantly, each continues to require some form of treatment to maintain a good level of functioning. Findings of a significant number of autoimmune disorders and laboratory markers of immune activation in this population may guide new diagnostic and treatment opportunities.Keywords: lorazepam, electroconvulsive therapy, dextromethorphan/quinidine, benzodiazepines, Trisomy 21, Bush-Francis Catatonia Rating ScaleMiles JHTakahashi NMuckerman JNowell KPIthman MDove Medical PressarticleLorazepamElectroconvulsive TherapyDextromethorphan/quinidineBenzodiazepinesTrisomy 21Bush-Francis Catatonia Rating Scale.Neurosciences. Biological psychiatry. NeuropsychiatryRC321-571Neurology. Diseases of the nervous systemRC346-429ENNeuropsychiatric Disease and Treatment, Vol Volume 15, Pp 2723-2741 (2019)
institution	DOAJ
collection	DOAJ
language	EN
topic	Lorazepam Electroconvulsive Therapy Dextromethorphan/quinidine Benzodiazepines Trisomy 21 Bush-Francis Catatonia Rating Scale. Neurosciences. Biological psychiatry. Neuropsychiatry RC321-571 Neurology. Diseases of the nervous system RC346-429
spellingShingle	Lorazepam Electroconvulsive Therapy Dextromethorphan/quinidine Benzodiazepines Trisomy 21 Bush-Francis Catatonia Rating Scale. Neurosciences. Biological psychiatry. Neuropsychiatry RC321-571 Neurology. Diseases of the nervous system RC346-429 Miles JH Takahashi N Muckerman J Nowell KP Ithman M Catatonia in Down syndrome: systematic approach to diagnosis, treatment and outcome assessment based on a case series of seven patients
description	Judith H Miles,1,2 Nicole Takahashi,2 Julie Muckerman,2 Kerri P Nowell,2,3 Muaid Ithman4 1Department of Child Health, University of Missouri Healthcare, Columbia, MO, USA; 2Thompson Center for Autism and Neurodevelopmental Disorders, University of Missouri, Columbia, MO, USA; 3Department of Health Psychology, University of Missouri Healthcare, Columbia, MO, USA; 4Department of Psychiatry, University of Missouri Health Care, Columbia, MO, USACorrespondence: Judith H MilesThompson Center for Autism and Neurodevelopmental Disorders, 205 Portland Street, Columbia, MO 65211, USATel +1 573 884 6838Fax +1 573 884 1151Email milesjh@missouri.eduObjective: The goal is to expand our knowledge of catatonia occurring in adolescents and young adults with Down syndrome (DS) by describing the first prospective, consecutive, well-characterized cohort of seven young people with DS diagnosed with catatonia and treated between 2013 and 2018, and to assess each patient’s treatment responses. Longitudinal assessment of each patient’s response to treatment is intended to provide clinicians and psychiatrists a firm foundation from which assess treatment efficacy.Study design: Young adults with Down syndrome were consecutively enrolled in the study as they were diagnosed with catatonia. A comprehensive data set included medical, laboratory, developmental, demographic, family, social and genetic data, including query into disorders for which individuals with DS are at risk. Catatonia was diagnosed based on an unequivocal history of regression, positive Bush-Francis Catatonia Rating Scale and positive response to intravenous lorazepam. Patients’ longitudinal progress was monitored using the Catatonia Impact Scale (CIS) developed for this purpose.Results: Seven consecutive DS patients, who presented with unequivocal regression were diagnosed with catatonia and treated for 2.7–6 years using standard-of-care therapies; primarily GABA agonist, lorazepam, electroconvulsive therapy (ECT) and glutamate antagonists (dextromethorphan/quinidine, memantine, minocycline). Responses to each treatment modality were assessed at clinic visits and through weekly electronic CIS reports.Conclusion: Seven young adults with DS were diagnosed with catatonia; all responded to Lorazepam and/or ECT therapy with good to very good results. Though ECT most dramatically returned patients to baseline, symptoms often returned requiring additional ECT. Dextromethorphan/quinidine, not used until mid-2017, appeared to reduce the reoccurrence of symptoms following ECT. Though all seven patients improved significantly, each continues to require some form of treatment to maintain a good level of functioning. Findings of a significant number of autoimmune disorders and laboratory markers of immune activation in this population may guide new diagnostic and treatment opportunities.Keywords: lorazepam, electroconvulsive therapy, dextromethorphan/quinidine, benzodiazepines, Trisomy 21, Bush-Francis Catatonia Rating Scale
format	article
author	Miles JH Takahashi N Muckerman J Nowell KP Ithman M
author_facet	Miles JH Takahashi N Muckerman J Nowell KP Ithman M
author_sort	Miles JH
title	Catatonia in Down syndrome: systematic approach to diagnosis, treatment and outcome assessment based on a case series of seven patients
title_short	Catatonia in Down syndrome: systematic approach to diagnosis, treatment and outcome assessment based on a case series of seven patients
title_full	Catatonia in Down syndrome: systematic approach to diagnosis, treatment and outcome assessment based on a case series of seven patients
title_fullStr	Catatonia in Down syndrome: systematic approach to diagnosis, treatment and outcome assessment based on a case series of seven patients
title_full_unstemmed	Catatonia in Down syndrome: systematic approach to diagnosis, treatment and outcome assessment based on a case series of seven patients
title_sort	catatonia in down syndrome: systematic approach to diagnosis, treatment and outcome assessment based on a case series of seven patients
publisher	Dove Medical Press
publishDate	2019
url	https://doaj.org/article/eb1875e19a184d488522e2a0c856e362
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_version_	1718401403117895680

Catatonia in Down syndrome: systematic approach to diagnosis, treatment and outcome assessment based on a case series of seven patients

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