Evidence map›Paper›PMID 31595705›Full record

ArticleMolecular genetics & genomic medicine2019

Targeted sequencing identifies novel variants in common and rare MODY genes.

Lucas S de Santana, Lilian A Caetano, Aline D Costa-Riquetto, Pedro C Franco, Renata P Dotto, André F Reis, Letícia S Weinert, Sandra P Silveiro, Marcio F Vendramini, Flaviene A do Prado and 9 more

Abstract read
In one paragraph

Article in Molecular genetics & genomic medicine, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 20 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
20citing papers in PubMed, 1 pooled it
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

20 citing papers in PubMed, 1 synthesis or guideline pooled it.

  1. Clinical and Genetic Characteristics ofJournal of diabetes research · 2021
    Pooled it
  2. Article
  3. Sulphonylurea efficacy and end-organ outcomes in the management of HNF4A-MODY.Diabetic medicine : a journal of the British Diabetic Association · 2025
    Article
  4. Article
  5. Review
  6. Autosomal Dominant, Long-Standing Dysglycemia in 2 Families with Unique Phenotypic Features.Clinical medicine insights. Endocrinology and diabetes · 2024
    Article
  7. Article
  8. Article
  9. Article
  10. Article
  11. Review
  12. Article
  13. Article
  14. Article
  15. Article
  16. Article
  17. Maturity Onset Diabetes of the Young-New Approaches for Disease Modelling.International journal of molecular sciences · 2021
    Review
  18. Article
  19. Review
  20. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

19 authors.

Lucas S de SantanaMonogenic Diabetes Group, Genetic Endocrinology Unit and Laboratory of Molecular & Cellular Endocrinology/LIM25, School of Medicine, University of Sao Paulo (USP), Sao Paulo, SP, Brazil.ORCID 0000-0003-0428-2386
Lilian A CaetanoMonogenic Diabetes Group, Genetic Endocrinology Unit and Laboratory of Molecular & Cellular Endocrinology/LIM25, School of Medicine, University of Sao Paulo (USP), Sao Paulo, SP, Brazil.ORCID 0000-0002-4645-5858
Aline D Costa-RiquettoMonogenic Diabetes Group, Genetic Endocrinology Unit and Laboratory of Molecular & Cellular Endocrinology/LIM25, School of Medicine, University of Sao Paulo (USP), Sao Paulo, SP, Brazil.
Pedro C FrancoMonogenic Diabetes Group, Genetic Endocrinology Unit and Laboratory of Molecular & Cellular Endocrinology/LIM25, School of Medicine, University of Sao Paulo (USP), Sao Paulo, SP, Brazil.
Renata P DottoDepartamento de Medicina, Disciplina de Endocrinologia, Universidade Federal de São Paulo (UNIFESP), Sao Paulo, SP, Brazil.
André F ReisDepartamento de Medicina, Disciplina de Endocrinologia, Universidade Federal de São Paulo (UNIFESP), Sao Paulo, SP, Brazil.
Letícia S WeinertUniversidade Federal do Rio Grande do Sul (UFRGS), Porto Alegre, RS, Brazil.
Sandra P SilveiroUniversidade Federal do Rio Grande do Sul (UFRGS), Porto Alegre, RS, Brazil.
Marcio F VendraminiServiço de Endocrinologia, Hospital do Servidor Público Estadual de São Paulo (HSPE-SP), Sao Paulo, SP, Brazil.
Flaviene A do PradoHospital Regional de Taguatinga da Secretaria de Saúde do Distrito Federal, Taguatinga, DF, Brazil.
Giovanna C P AbrahãoPontifícia Universidade Católica de São Paulo (PUCSP), Sao Paulo, SP, Brazil.
Ana Gregória F P de AlmeidaInstituto Federal de Educação, Ciência e Tecnologia do Maranhão (IFMA), Sao Luis, MA, Brazil.
Maria da G Rodrigues TavaresServiço de Endocrinologia do Hospital Universitário, Universidade Federal do Maranhão (UFMA), Sao Luis, MA, Brazil.
Wagner Rodrigo B GonçalvesHospital do Servidor Público Municipal de São Paulo (HSPM-SP), Sao Paulo, SP, Brazil.
Augusto C Santomauro JuniorServiço de Endocrinologia Prof. Dr. Fadlo Fraige Filho, Hospital Beneficência Portuguesa de São Paulo (BP-SP), Sao Paulo, SP, Brazil.
Bruno HalpernDepartamento de Endocrinologia e Metabologia, Hospital das Clínicas, Faculdade de Medicina, Universidade de São Paulo (USP), Sao Paulo, SP, Brazil.
Alexander A L JorgeMonogenic Diabetes Group, Genetic Endocrinology Unit and Laboratory of Molecular & Cellular Endocrinology/LIM25, School of Medicine, University of Sao Paulo (USP), Sao Paulo, SP, Brazil.ORCID 0000-0003-2567-7360
Marcia NeryDiabetes Unit, Clinics Hospital, School of Medicine, University of Sao Paulo (USP), Sao Paulo, SP, Brazil.
Milena G TelesMonogenic Diabetes Group, Genetic Endocrinology Unit and Laboratory of Molecular & Cellular Endocrinology/LIM25, School of Medicine, University of Sao Paulo (USP), Sao Paulo, SP, Brazil.ORCID 0000-0002-0303-5335

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundMaturity-onset diabetes of the young (MODY) is a form of monogenic diabetes with autosomal dominant inheritance. To date, mutations in 11 genes have been frequently associated with this phenotype. In Brazil, few cohorts have been screened for MODY, all using a candidate gene approach, with a high prevalence of undiagnosed cases (MODY-X).

methodsWe conducted a next-generation sequencing target panel (tNGS) study to investigate, for the first time, a Brazilian cohort of MODY patients with a negative prior genetic analysis. One hundred and two patients were selected, of which 26 had an initial clinical suspicion of MODY-GCK and 76 were non-GCK MODY.

resultsAfter excluding all benign and likely benign variants and variants of uncertain significance, we were able to assign a genetic cause for 12.7% (13/102) of the probands. Three rare MODY subtypes were identified (PDX1/NEUROD1/ABCC8), and eight variants had not been previously described/mapped in genomic databases. Important clinical findings were evidenced in some cases after genetic diagnosis, such as MODY-PDX1/HNF1B.

conclusionA multiloci genetic approach allowed the identification of rare MODY subtypes, reducing the large percentage of MODY-X in Brazilian cases and contributing to a better clinical, therapeutic, and prognostic characterization of these rare phenotypes.

Indexed as

AdolescentAdultBasic Helix-Loop-Helix ProteinsBrazilCohort StudiesDiabetes Mellitus, Type 2FemaleGenetic Predisposition to DiseaseGenetic TestingHigh-Throughput Nucleotide SequencingHomeodomain ProteinsHumansMaleSequence Analysis, DNASulfonylurea ReceptorsTrans-ActivatorsABCC8 protein, humanBasic Helix-Loop-Helix ProteinsHomeodomain ProteinsNEUROD1 protein, humanpancreatic and duodenal homeobox 1 proteinSulfonylurea ReceptorsTrans-ActivatorsACMG/AMPMODYMODY-Xtargeted sequencing

Identifiers

PMID31595705
PMCPMC6900361

What Socratic holds

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Registered trials

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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 Socratic graph.