Evidence map›Paper›PMID 38721574›Full record

ArticleJournal of pediatric genetics2024

Identifying Genetic Etiology in Patients with Intellectual Disability: An Experience in Public Health Services in Northeastern Brazil.

Acacia Fernandes Lacerda de Carvalho, Esmeralda Santos Alves, Paula Monique Leite Pitanga, Erlane Marques Ribeiro, Maria Juliana Rodovalho Doriqui, Maria Betânia Pereira Toralles, Bianca Arcaro Topázio, Jéssica Fernandes Dos Santos, Renata Lúcia Leite Ferreira de Lima, Leslie Domenici Kulikowski and 1 more

Abstract read
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Article in Journal of pediatric genetics, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

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1 · What the graph read from it

What it found

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2 · The registry

The trial behind it

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3 · Its place in the literature

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4 · The record

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5 · Who and what money

Authors and funding

11 authors.

Acacia Fernandes Lacerda de CarvalhoLaboratory of Human Genetics and Mutagenesis, Institute of Biology, Federal University Bahia (UFBA), Salvador, Bahia, Brazil.ORCID 0000-0003-3639-338X
Esmeralda Santos AlvesLaboratory of Human Genetics and Mutagenesis, Institute of Biology, Federal University Bahia (UFBA), Salvador, Bahia, Brazil.ORCID 0000-0002-9912-8404
Paula Monique Leite PitangaLaboratory of Human Genetics and Mutagenesis, Institute of Biology, Federal University Bahia (UFBA), Salvador, Bahia, Brazil.
Erlane Marques RibeiroFaculty of Medicine Estacio of Juazeiro Norte, Estacio-FMJ, Hospital Infantil Albert Sabin, Fortaleza, Ceará, Brazil.
Maria Juliana Rodovalho DoriquiAssociation of Parents and Friends of Exceptional Children (APAE), São Luiz, Maranhão, Brazil.
Maria Betânia Pereira TorallesMedical School of Medicine, Medical Genetic Service - Edgard Santos Teaching Hospital/Federal University of Bahia, Salvador, Bahia, Brazil.
Bianca Arcaro TopázioLaboratory of Human Genetics and Mutagenesis, Institute of Biology, Federal University Bahia (UFBA), Salvador, Bahia, Brazil.
Jéssica Fernandes Dos SantosLaboratory of Human Genetics and Mutagenesis, Institute of Biology, Federal University Bahia (UFBA), Salvador, Bahia, Brazil.
Renata Lúcia Leite Ferreira de LimaLaboratory of Human Genetics and Mutagenesis, Institute of Biology, Federal University Bahia (UFBA), Salvador, Bahia, Brazil.
Leslie Domenici KulikowskiDepartment of Pathology, Cytogenomics Laboratory - LIM 03, University of São Paulo, São Paulo, Brazil.
Angelina Xavier AcostaMedical School of Medicine, Medical Genetic Service - Edgard Santos Teaching Hospital/Federal University of Bahia, Salvador, Bahia, Brazil.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Intellectual disability (ID) is considered a common neuropsychiatric disorder that affects up to 3% of the population. The etiologic origin of ID may be genetic, environmental, and multifactorial. Chromosomopathies are relatively common among the genetic causes of ID, especially in the most severe cases and those associated with dysmorphic features. Currently, the application of new molecular cytogenetics technologies has increasingly allowed the identification of microdeletions, microduplications, and unbalanced translocations as causes of ID. The objective of this study was to investigate the etiology of ID in patients admitted to a public hospital in Northeastern Brazil. In total, 119 patients with ID who had normal karyotypes and fragile X exams participated in this study. The patients were initially physically examined for microdeletion syndromes and then tested using fluorescence in situ hybridization (FISH), multiplex ligation-dependent probe amplification (MLPA), methylation-sensitive polymerase chain reaction (MS-PCR), and chromosome microarray analysis (CMA), according to clinical suspicion. Patients with no diagnoses after FISH, MLPA, and/or MS-PCR evaluations were subsequently tested by CMA. The rate of etiologic diagnoses of ID in the current study was 28%. FISH diagnosed 25 out of 79 tested (31%), MLPA diagnosed 26 out of 79 tested (32%), MS-PCR diagnosed 7 out of 20 tested (35%), and the single nucleotide polymorphism array diagnosed 6 out of 27 tested (22%). Although the CMA is the most complete and recommended tool for the diagnosis of microdeletions, microduplications, and unbalance translocations in patients with ID, FISH, MLPA, and MS-PCR testing can be used as the first tests for specific syndromes, as long as the patients are first physically screened clinically, especially in the public health networks system in Brazil, where resources are scarce.

Indexed as

chromosome microarray analysiscopy number variationdysmorphismFISHintellectual disability

Identifiers

PMID38721574
PMCPMC11076089

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.