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Clinical and Genetic Aspects of Phelan-McDermid Syndrome: An Interdisciplinary Approach to Management.
Cammarata-Scalisi, Francisco; Callea, Michele; Martinelli, Diego; Willoughby, Colin Eric; Tadich, Antonio Cárdenas; Araya Castillo, Maykol; Lacruz-Rengel, María Angelina; Medina, Marco; Grimaldi, Piercesare; Bertini, Enrico; Nevado, Julián.
Afiliação
  • Cammarata-Scalisi F; Pediatric Service, Regional of Antofagasta Hospital, Antofagasta 1240835, Chile.
  • Callea M; Pediatric Dentistry and Special Dental Care Unit, Meyer Children's University Hospital, 50139 Florence, Italy.
  • Martinelli D; Unit of Metabolism, Bambino Gesù Children's Research Hospital IRCCS, 00165 Rome, Italy.
  • Willoughby CE; Genomic Medicine, Biomedical Sciences Research Institute, Ulster University, Coleraine Campus, Coleraine BT52 1SA, Northern Ireland, UK.
  • Tadich AC; Pediatric Service, Regional of Antofagasta Hospital, Antofagasta 1240835, Chile.
  • Araya Castillo M; Clinical Laboratory, Regional of Antofagasta Hospital, Antofagasta 1240835, Chile.
  • Lacruz-Rengel MA; Neuropediatrics Service, Childcare and Pediatric Department, University of Los Andes, Mérida 51101, Venezuela.
  • Medina M; Pediatric Service, Regional of Antofagasta Hospital, Antofagasta 1240835, Chile.
  • Grimaldi P; Department of Public Health and Pediatric Sciences, University of Torino, 10126 Torino, Italy.
  • Bertini E; Unit of Neuromuscular and Neurodegenerative Disorders, Laboratory of Molecular Medicine, Department of Neurosciences, Bambino Gesu' Children's Research Hospital IRCCS, 00165 Rome, Italy.
  • Nevado J; Instituto de Genética Médica y Molecular (INGEMM), Instituto de Investigación del Hospital Universitario La Paz (IdIPaz), 28046 Madrid, Spain.
Genes (Basel) ; 13(3)2022 03 12.
Article em En | MEDLINE | ID: mdl-35328058
Phelan-McDermid syndrome (PMS) is a rare, heterogeneous, and complex neurodevelopmental disorder. It is generally caused by a heterozygous microdeletion of contiguous genes located in the distal portion of the long arm of chromosome 22, including the SHANK3 gene. Sequence variants of SHANK3, including frameshift, nonsense mutations, small indels and splice site mutations also result in PMS. Furthermore, haploinsufficiency in SHANK3 has been suggested as the main cause of PMS. SHANK3 is also associated with intellectual disability, autism spectrum disorder and schizophrenia. The phenotype of PMS is variable, and lacks a distinctive phenotypic characteristic, so the clinical diagnosis should be confirmed by genetic analysis. PMS is a multi-system disorder, and clinical care must encompass various specialties and therapists. The role of risperidone, intranasal insulin, insulin growth factor 1, and oxytocin as potential therapeutic options in PMS will be discussed in this review. The diagnosis of PMS is important to provide an appropriate clinical evaluation, treatment, and genetic counseling.
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Texto completo: 1 Base de dados: MEDLINE Assunto principal: Transtornos Cromossômicos / Transtorno do Espectro Autista Tipo de estudo: Diagnostic_studies Limite: Humans Idioma: En Ano de publicação: 2022 Tipo de documento: Article

Texto completo: 1 Base de dados: MEDLINE Assunto principal: Transtornos Cromossômicos / Transtorno do Espectro Autista Tipo de estudo: Diagnostic_studies Limite: Humans Idioma: En Ano de publicação: 2022 Tipo de documento: Article