A causality algorithm to guide diagnosis and treatment of catatonia due to autoimmune conditions in children and adolescents

Pediatric catatonia is a rare and life-threatening syndrome. Around 20% of juvenile catatonia is associated with organic condition (Consoli et al., 2012). Autoimmune conditions represent a diagnostic and therapeutic challenge since specific antibodies can be missed. To facilitate decision making, we...

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Vydáno v:Schizophrenia research Ročník 200; s. 68 - 76
Hlavní autoři: Ferrafiat, Vladimir, Raffin, Marie, Freri, Elena, Granata, Tiziana, Nardocci, Nardo, Zibordi, Federica, Bodeau, Nicolas, Benarous, Xavier, Olliac, Bertrand, Riquin, Elise, Xavier, Jean, Viaux, Sylvie, Haroche, Julien, Amoura, Zahir, Gerardin, Priscille, Cohen, David, Consoli, Angèle
Médium: Journal Article
Jazyk:angličtina
Vydáno: Netherlands Elsevier B.V 01.10.2018
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ISSN:0920-9964, 1573-2509, 1573-2509
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Abstract Pediatric catatonia is a rare and life-threatening syndrome. Around 20% of juvenile catatonia is associated with organic condition (Consoli et al., 2012). Autoimmune conditions represent a diagnostic and therapeutic challenge since specific antibodies can be missed. To facilitate decision making, we recently formulated a causality assessment score (CAUS) using a stepwise approach and an immunosuppressive therapeutic challenge (Ferrafiat et al., 2016). Our objectives were to validate retrospectively CAUS and to define its threshold for an accurate distinction between organic catatonia and non-organic catatonia, and specifically between autoimmune catatonia and non-organic catatonia. To obtain a sufficient number of cases with organic catatonia, we pooled two samples (N=104) – one from a child psychiatry center, the other from neuro-pediatrics center – expert in catatonia and autoimmune conditions. Organic conditions were diagnosed using a multidisciplinary approach and numerous paraclinical investigations. Given the binary classification needs, we used receiver operating characteristic (ROC) analysis (Peacock and Peacock, 2010) to calculate the best classification threshold. The cohort included 67 cases of non-organic catatonia and 37 cases of organic catatonia. ROC analysis showed that the CAUS performance in discriminating both organic catatonia vs. non-organic catatonia, and autoimmune catatonia vs. non-organic catatonia was excellent (Area Under the Curve=0.99). In both analyses, for a CAUS threshold≥5, accuracy equaled to 0.96. Regarding juvenile catatonia, the use of the CAUS score algorithm combining a therapeutic challenge and a threshold≥5 may help to diagnose and treat autoimmune conditions even without formal identification of auto-antibodies.
AbstractList Pediatric catatonia is a rare and life-threatening syndrome. Around 20% of juvenile catatonia is associated with organic condition (Consoli et al., 2012). Autoimmune conditions represent a diagnostic and therapeutic challenge since specific antibodies can be missed. To facilitate decision making, we recently formulated a causality assessment score (CAUS) using a stepwise approach and an immunosuppressive therapeutic challenge (Ferrafiat et al., 2016). Our objectives were to validate retrospectively CAUS and to define its threshold for an accurate distinction between organic catatonia and non-organic catatonia, and specifically between autoimmune catatonia and non-organic catatonia. To obtain a sufficient number of cases with organic catatonia, we pooled two samples (N=104) – one from a child psychiatry center, the other from neuro-pediatrics center – expert in catatonia and autoimmune conditions. Organic conditions were diagnosed using a multidisciplinary approach and numerous paraclinical investigations. Given the binary classification needs, we used receiver operating characteristic (ROC) analysis (Peacock and Peacock, 2010) to calculate the best classification threshold. The cohort included 67 cases of non-organic catatonia and 37 cases of organic catatonia. ROC analysis showed that the CAUS performance in discriminating both organic catatonia vs. non-organic catatonia, and autoimmune catatonia vs. non-organic catatonia was excellent (Area Under the Curve=0.99). In both analyses, for a CAUS threshold≥5, accuracy equaled to 0.96. Regarding juvenile catatonia, the use of the CAUS score algorithm combining a therapeutic challenge and a threshold≥5 may help to diagnose and treat autoimmune conditions even without formal identification of auto-antibodies.
Pediatric catatonia is a rare and life-threatening syndrome. Around 20% of juvenile catatonia is associated with organic condition (Consoli et al., 2012). Autoimmune conditions represent a diagnostic and therapeutic challenge since specific antibodies can be missed. To facilitate decision making, we recently formulated a causality assessment score (CAUS) using a stepwise approach and an immunosuppressive therapeutic challenge (Ferrafiat et al., 2016). Our objectives were to validate retrospectively CAUS and to define its threshold for an accurate distinction between organic catatonia and non-organic catatonia, and specifically between autoimmune catatonia and non-organic catatonia.OBJECTIVESPediatric catatonia is a rare and life-threatening syndrome. Around 20% of juvenile catatonia is associated with organic condition (Consoli et al., 2012). Autoimmune conditions represent a diagnostic and therapeutic challenge since specific antibodies can be missed. To facilitate decision making, we recently formulated a causality assessment score (CAUS) using a stepwise approach and an immunosuppressive therapeutic challenge (Ferrafiat et al., 2016). Our objectives were to validate retrospectively CAUS and to define its threshold for an accurate distinction between organic catatonia and non-organic catatonia, and specifically between autoimmune catatonia and non-organic catatonia.To obtain a sufficient number of cases with organic catatonia, we pooled two samples (N=104) - one from a child psychiatry center, the other from neuro-pediatrics center - expert in catatonia and autoimmune conditions. Organic conditions were diagnosed using a multidisciplinary approach and numerous paraclinical investigations. Given the binary classification needs, we used receiver operating characteristic (ROC) analysis (Peacock and Peacock, 2010) to calculate the best classification threshold.METHODTo obtain a sufficient number of cases with organic catatonia, we pooled two samples (N=104) - one from a child psychiatry center, the other from neuro-pediatrics center - expert in catatonia and autoimmune conditions. Organic conditions were diagnosed using a multidisciplinary approach and numerous paraclinical investigations. Given the binary classification needs, we used receiver operating characteristic (ROC) analysis (Peacock and Peacock, 2010) to calculate the best classification threshold.The cohort included 67 cases of non-organic catatonia and 37 cases of organic catatonia. ROC analysis showed that the CAUS performance in discriminating both organic catatonia vs. non-organic catatonia, and autoimmune catatonia vs. non-organic catatonia was excellent (Area Under the Curve=0.99). In both analyses, for a CAUS threshold≥5, accuracy equaled to 0.96.RESULTSThe cohort included 67 cases of non-organic catatonia and 37 cases of organic catatonia. ROC analysis showed that the CAUS performance in discriminating both organic catatonia vs. non-organic catatonia, and autoimmune catatonia vs. non-organic catatonia was excellent (Area Under the Curve=0.99). In both analyses, for a CAUS threshold≥5, accuracy equaled to 0.96.Regarding juvenile catatonia, the use of the CAUS score algorithm combining a therapeutic challenge and a threshold≥5 may help to diagnose and treat autoimmune conditions even without formal identification of auto-antibodies.CONCLUSIONRegarding juvenile catatonia, the use of the CAUS score algorithm combining a therapeutic challenge and a threshold≥5 may help to diagnose and treat autoimmune conditions even without formal identification of auto-antibodies.
Author Gerardin, Priscille
Cohen, David
Haroche, Julien
Raffin, Marie
Ferrafiat, Vladimir
Freri, Elena
Amoura, Zahir
Granata, Tiziana
Benarous, Xavier
Olliac, Bertrand
Riquin, Elise
Viaux, Sylvie
Consoli, Angèle
Xavier, Jean
Zibordi, Federica
Bodeau, Nicolas
Nardocci, Nardo
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  orcidid: 0000-0002-9285-1034
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  fullname: Ferrafiat, Vladimir
  email: Vladimir.Ferrafiat@chu-rouen.fr
  organization: Department of Child and Adolescent Psychiatry, Université Pierre et Marie Curie, Hôpital Pitié-Salpêtrière, AP-HP, 47-83 Boulevard de l'Hôpital, 75013 Paris, France
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  organization: Department of Child and Adolescent Psychiatry, Université Pierre et Marie Curie, Hôpital Pitié-Salpêtrière, AP-HP, 47-83 Boulevard de l'Hôpital, 75013 Paris, France
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  givenname: Elena
  surname: Freri
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  organization: Department of Pediatric Neuroscience, Foundation I.R.C.C.S. Neurological Institute “C. Besta”, Via Celoria 11, 20133 Milan, Italy
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  surname: Zibordi
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  organization: Department of Pediatric Neuroscience, Foundation I.R.C.C.S. Neurological Institute “C. Besta”, Via Celoria 11, 20133 Milan, Italy
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  givenname: Nicolas
  surname: Bodeau
  fullname: Bodeau, Nicolas
  organization: Department of Child and Adolescent Psychiatry, Université Pierre et Marie Curie, Hôpital Pitié-Salpêtrière, AP-HP, 47-83 Boulevard de l'Hôpital, 75013 Paris, France
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  givenname: Elise
  surname: Riquin
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  organization: Department of Child and Adolescent Psychiatry, Université Pierre et Marie Curie, Hôpital Pitié-Salpêtrière, AP-HP, 47-83 Boulevard de l'Hôpital, 75013 Paris, France
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  givenname: Sylvie
  surname: Viaux
  fullname: Viaux, Sylvie
  organization: Department of Child and Adolescent Psychiatry, Université Pierre et Marie Curie, Hôpital Pitié-Salpêtrière, AP-HP, 47-83 Boulevard de l'Hôpital, 75013 Paris, France
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  givenname: Julien
  surname: Haroche
  fullname: Haroche, Julien
  organization: French National Reference Center for Rare Systemic AutoImmune Disorders, E3M Institute, Université Pierre et Marie Curie, Hôpital Pitié-Salpêtrière, AP-HP, 47-83 Boulevard de l'Hôpital, 75013 Paris, France
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  givenname: Zahir
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  surname: Gerardin
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  surname: Cohen
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  givenname: Angèle
  surname: Consoli
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  organization: Department of Child and Adolescent Psychiatry, Université Pierre et Marie Curie, Hôpital Pitié-Salpêtrière, AP-HP, 47-83 Boulevard de l'Hôpital, 75013 Paris, France
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Keywords Encephalitis
Catatonia
Autoimmune condition
Immunosuppressive treatment
Language English
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Snippet Pediatric catatonia is a rare and life-threatening syndrome. Around 20% of juvenile catatonia is associated with organic condition (Consoli et al., 2012)....
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SubjectTerms Autoimmune condition
Catatonia
Encephalitis
Immunosuppressive treatment
Title A causality algorithm to guide diagnosis and treatment of catatonia due to autoimmune conditions in children and adolescents
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https://dx.doi.org/10.1016/j.schres.2017.06.036
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