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A Novel Analog Reasoning Paradigm: New Insights in Intellectually Disabled Patients.
Curie, Aurore; Brun, Amandine; Cheylus, Anne; Reboul, Anne; Nazir, Tatjana; Bussy, Gérald; Delange, Karine; Paulignan, Yves; Mercier, Sandra; David, Albert; Marignier, Stéphanie; Merle, Lydie; de Fréminville, Bénédicte; Prieur, Fabienne; Till, Michel; Mortemousque, Isabelle; Toutain, Annick; Bieth, Eric; Touraine, Renaud; Sanlaville, Damien; Chelly, Jamel; Kong, Jian; Ott, Daniel; Kassai, Behrouz; Hadjikhani, Nouchine; Gollub, Randy L; des Portes, Vincent.
  • Curie A; Centre de Référence, Déficiences Intellectuelles de Causes Rares, Hôpital Femme Mère Enfant, Hospices Civils de Lyon, Bron, France.
  • Brun A; CNRS UMR 5304, L2C2, Institut des Sciences Cognitives, Bron, France.
  • Cheylus A; Université de Lyon, Faculté de médecine Lyon Sud-Charles Mérieux, Lyon, France.
  • Reboul A; Athinoula A. Martinos Center for Biomedical Imaging, Massachusetts General Hospital, Charlestown, MA, United States of America.
  • Nazir T; EPICIME-CIC 1407/Inserm, UMR5558, LBBE, Université de Lyon, France.
  • Bussy G; Centre de Référence, Déficiences Intellectuelles de Causes Rares, Hôpital Femme Mère Enfant, Hospices Civils de Lyon, Bron, France.
  • Delange K; CNRS UMR 5304, L2C2, Institut des Sciences Cognitives, Bron, France.
  • Paulignan Y; CNRS UMR 5304, L2C2, Institut des Sciences Cognitives, Bron, France.
  • Mercier S; CNRS UMR 5304, L2C2, Institut des Sciences Cognitives, Bron, France.
  • David A; CNRS UMR 5304, L2C2, Institut des Sciences Cognitives, Bron, France.
  • Marignier S; Centre de Référence, Déficiences Intellectuelles de Causes Rares, Hôpital Femme Mère Enfant, Hospices Civils de Lyon, Bron, France.
  • Merle L; CNRS UMR 5304, L2C2, Institut des Sciences Cognitives, Bron, France.
  • de Fréminville B; Centre de Référence, Déficiences Intellectuelles de Causes Rares, Hôpital Femme Mère Enfant, Hospices Civils de Lyon, Bron, France.
  • Prieur F; CNRS UMR 5304, L2C2, Institut des Sciences Cognitives, Bron, France.
  • Till M; CNRS UMR 5304, L2C2, Institut des Sciences Cognitives, Bron, France.
  • Mortemousque I; Génétique, Centre Hospitalier Universitaire, Nantes, France.
  • Toutain A; Génétique, Centre Hospitalier Universitaire, Nantes, France.
  • Bieth E; Centre de Référence, Déficiences Intellectuelles de Causes Rares, Hôpital Femme Mère Enfant, Hospices Civils de Lyon, Bron, France.
  • Touraine R; Centre de Recherche en Neurosciences de Lyon, INSERM U1028, CNRS UMR5292, CH le Vinatier, France.
  • Sanlaville D; Génétique, Centre Hospitalier Universitaire, Saint-Etienne, France.
  • Chelly J; Génétique, Centre Hospitalier Universitaire, Saint-Etienne, France.
  • Kong J; Médecine interne, Hôpital Saint Luc Saint Joseph, Lyon, France.
  • Ott D; Génétique, Centre Hospitalier Universitaire, Tours, France.
  • Kassai B; Génétique, Centre Hospitalier Universitaire, Tours, France.
  • Hadjikhani N; Génétique, Centre Hospitalier Universitaire, Toulouse, France.
  • Gollub RL; Génétique, Centre Hospitalier Universitaire, Saint-Etienne, France.
  • des Portes V; Service de génétique, Hôpital Femme Mère Enfant, Hospices Civils de Lyon, CRNL, CNRS UMR5292, INSERM U1028, Université Claude Bernard Lyon I, Lyon, France.
PLoS One ; 11(2): e0149717, 2016.
Article en En | MEDLINE | ID: mdl-26918704
ABSTRACT

BACKGROUND:

Intellectual Disability (ID) is characterized by deficits in intellectual functions such as reasoning, problem-solving, planning, abstract thinking, judgment, and learning. As new avenues are emerging for treatment of genetically determined ID (such as Down's syndrome or Fragile X syndrome), it is necessary to identify objective reliable and sensitive outcome measures for use in clinical trials.

OBJECTIVE:

We developed a novel visual analogical reasoning paradigm, inspired by the Progressive Raven's Matrices, but appropriate for Intellectually Disabled patients. This new paradigm assesses reasoning and inhibition abilities in ID patients.

METHODS:

We performed behavioural analyses for this task (with a reaction time and error rate analysis, Study 1) in 96 healthy controls (adults and typically developed children older than 4) and 41 genetically determined ID patients (Fragile X syndrome, Down syndrome and ARX mutated patients). In order to establish and quantify the cognitive strategies used to solve the task, we also performed an eye-tracking analysis (Study 2).

RESULTS:

Down syndrome, ARX and Fragile X patients were significantly slower and made significantly more errors than chronological age-matched healthy controls. The effect of inhibition on error rate was greater than the matrix complexity effect in ID patients, opposite to findings in adult healthy controls. Interestingly, ID patients were more impaired by inhibition than mental age-matched healthy controls, but not by the matrix complexity. Eye-tracking analysis made it possible to identify the strategy used by the participants to solve the task. Adult healthy controls used a matrix-based strategy, whereas ID patients used a response-based strategy. Furthermore, etiologic-specific reasoning differences were evidenced between ID patients groups.

CONCLUSION:

We suggest that this paradigm, appropriate for ID patients and developmental populations as well as adult healthy controls, provides an objective and quantitative assessment of visual analogical reasoning and cognitive inhibition, enabling testing for the effect of pharmacological or behavioural intervention in these specific populations.
Asunto(s)

Texto completo: 1 Banco de datos: MEDLINE Asunto principal: Pensamiento / Discapacidad Intelectual Tipo de estudio: Observational_studies / Prognostic_studies / Risk_factors_studies Límite: Adolescent / Adult / Female / Humans / Male / Middle aged Idioma: En Año: 2016 Tipo del documento: Article

Texto completo: 1 Banco de datos: MEDLINE Asunto principal: Pensamiento / Discapacidad Intelectual Tipo de estudio: Observational_studies / Prognostic_studies / Risk_factors_studies Límite: Adolescent / Adult / Female / Humans / Male / Middle aged Idioma: En Año: 2016 Tipo del documento: Article