Vladimir Ferrafiat1, Marie Raffin2, Elena Freri3, Tiziana Granata3, Nardo Nardocci3, Federica Zibordi3, Nicolas Bodeau2, Xavier Benarous2, Bertrand Olliac2, Elise Riquin4, Jean Xavier2, Sylvie Viaux2, Julien Haroche5, Zahir Amoura5, Priscille Gerardin6, David Cohen7, Angèle Consoli8. 1. 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; Department of Child and Adolescent Psychiatry, Université de Rouen, Hôpital Charles Nicolle, 1 rue de Germont, 76000 Rouen, France. Electronic address: Vladimir.Ferrafiat@chu-rouen.fr. 2. 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. 3. Department of Pediatric Neuroscience, Foundation I.R.C.C.S. Neurological Institute "C. Besta", Via Celoria 11, 20133 Milan, Italy. 4. Pediatric Psychiatry Department, Hôpital Universitaire d'Angers, 4 Rue Larrey, 49100 Angers, France. 5. 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. 6. Department of Child and Adolescent Psychiatry, Université de Rouen, Hôpital Charles Nicolle, 1 rue de Germont, 76000 Rouen, France. 7. 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; CNRS UMR 7222, Institut des Systèmes Intelligents et Robotiques, Université Pierre et Marie Curie, Hôpital Pitié-Salpêtrière, AP-HP, 47-83 Boulevard de l'Hôpital, 75013 Paris, France. 8. 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; Groupe de Recherche Clinique, Abord dimensionnel des épisodes psychotiques de l'enfant et de l'adolescent: implications génétiques, métaboliques et auto-immunes, Université Pierre et Marie Curie, Hôpital Pitié-Salpêtrière, AP-HP, 47-83 Boulevard de l'Hôpital, 75013 Paris, France.
Abstract
OBJECTIVES: 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. METHOD: 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. RESULTS: 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. CONCLUSION: 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.
OBJECTIVES: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. METHOD: 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. RESULTS: 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. CONCLUSION: 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.
Authors: GenaLynne C Mooneyham; Vladimir Ferrafiat; Erin Stolte; D Catherine Fuchs; David Cohen Journal: Front Psychiatry Date: 2021-03-29 Impact factor: 4.157