Evidence map›Paper›PMID 35503492›Full record

ArticleJournal of clinical immunology2022

SCID and Other Inborn Errors of Immunity with Low TRECs - the Brazilian Experience.

Lucila Akune Barreiros, Jusley Lira Sousa, Christoph Geier, Alexander Leiss-Piller, Marilia Pylles Patto Kanegae, Tábata Takahashi França, Bertrand Boisson, Alessandra Miramontes Lima, Beatriz Tavares Costa-Carvalho, Carolina Sanchez Aranda and 20 more

Abstract read
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In one paragraph

Article in Journal of clinical immunology, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 12 papers, 2 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
12citing papers in PubMed, 2 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

12 citing papers in PubMed, 2 syntheses or guidelines pooled it.

  1. Pooled it
  2. Pooled it
  3. Article
  4. Article
  5. Article
  6. Newborn screening in Brazil: realities and challenges.Journal of community genetics · 2025
    Review
  7. Article
  8. Article
  9. Review
  10. First pilot study for newborn screening of severe T and B lymphopenias in ColombiaBiomedica : revista del Instituto Nacional de Salud · 2024
    Article
  11. Review
  12. Review
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

30 authors.

Lucila Akune BarreirosLaboratory of Human Immunology, Department of Immunology, Institute of Biomedical Sciences, University of São Paulo, 1730, Av. Professor Lineu Prestes, Sao Paulo, SP, 05508-000, Brazil.
Jusley Lira SousaLaboratory of Human Immunology, Department of Immunology, Institute of Biomedical Sciences, University of São Paulo, 1730, Av. Professor Lineu Prestes, Sao Paulo, SP, 05508-000, Brazil.
Christoph GeierImmunology Outpatient Clinic, Vienna, Austria.
Alexander Leiss-PillerImmunology Outpatient Clinic, Vienna, Austria.
Marilia Pylles Patto KanegaeLaboratory of Human Immunology, Department of Immunology, Institute of Biomedical Sciences, University of São Paulo, 1730, Av. Professor Lineu Prestes, Sao Paulo, SP, 05508-000, Brazil.
Tábata Takahashi FrançaLaboratory of Human Immunology, Department of Immunology, Institute of Biomedical Sciences, University of São Paulo, 1730, Av. Professor Lineu Prestes, Sao Paulo, SP, 05508-000, Brazil.
Bertrand BoissonSt. Giles Laboratory of Human Genetics of Infectious Diseases, Rockefeller Branch, The Rockefeller University, New York, NY, USA.
Alessandra Miramontes LimaSabará Hospital, PENSI Institute - Jose Luiz Egydio Setubal Foundation, Sao Paulo, SP, Brazil.
Beatriz Tavares Costa-CarvalhoUniversidade Federal de São Paulo, Sao Paulo, SP, Brazil.
Carolina Sanchez ArandaUniversidade Federal de São Paulo, Sao Paulo, SP, Brazil.
Maria Isabel de Moraes-PintoUniversidade Federal de São Paulo, Sao Paulo, SP, Brazil.
Gesmar Rodrigues Silva SegundoDepartamento de Pediatra, Universidade Federal de Uberlândia, Uberlandia, MG, Brazil.
Janaira Fernandes Severo FerreiraHospital Infantil Albert Sabin, Fortaleza, CE, Brazil.
Fabíola Scancetti TavaresHospital da Criança de Brasília José de Alencar, Brasilia, DF, Brazil.
Flávia Alice Timburiba de Medeiros GuimarãesHospital Materno Infantil de Brasília, Brasilia, DF, Brazil.
Eliana Cristina ToledoFaculdade de Medicina de São José Do Rio Preto, Sao Jose Do Rio Preto, SP, Brazil.
Ana Carolina da Matta AinDepartamento de Pediatria E Imunologia, Hospital Universitário de Taubaté, Universidade de Taubaté, Taubate, SP, Brazil.
Iramirton Figueirêdo MoreiraHospital Universitário Prof. Alberto Antunes, Maceio, AL, Brazil.
Gustavo SoldatelliHospital das Clínicas, Universidade Federal de Santa Caratina, Florianopolis, SC, Brazil.
Anete Sevciovic GrumachFaculdade de Medicina, Centro Universitário FMABC, Santo Andre, SP, Brazil.
Mayra de Barros DornaInstituto da Criança, Hospital das Clínicas, Faculdade de Medicina da Universidade de São Paulo, Sao Paulo, SP, Brazil.
Cristina Worm WeberHospital das Clínicas de Porto Alegre, Porto Alegre, RS, Brazil.
Regina Sumiko Watanabe Di GesuHospital da Criança Conceição, Porto Alegre, RS, Brazil.
Vera Maria DantasDepartamento de Pediatria, Universidade Federal Do Rio Grande Do Norte, Natal, RN, Brazil.
Fátima Rodrigues FernandesSabará Hospital, PENSI Institute - Jose Luiz Egydio Setubal Foundation, Sao Paulo, SP, Brazil.
Troy Robert TorgersonAllen Institute for Immunology, Seattle, USA.
Hans Dietrich OchsDepartment of Pediatrics, University of Washington School of Medicine and Seattle Children's Research Institute, Seattle, USA.
Jacinta BustamanteSt. Giles Laboratory of Human Genetics of Infectious Diseases, Rockefeller Branch, The Rockefeller University, New York, NY, USA.
Jolan Eszter WalterUniversity of South Florida at Johns Hopkins All Children's Hospital, Saint Petersburg, FL, USA.
Antonio Condino-NetoLaboratory of Human Immunology, Department of Immunology, Institute of Biomedical Sciences, University of São Paulo, 1730, Av. Professor Lineu Prestes, Sao Paulo, SP, 05508-000, Brazil. antoniocondino@gmail.com.ORCID 0000-0002-1069-3117

Funding

Centro Nacional de Desenvolvimento Científico e Tecnológico 301299/2018-8Fundação de Amparo à Pesquisa do Estado de São Paulo 2016/22158-3Fundação de Amparo à Pesquisa do Estado de São Paulo 2018/09407-0Ministério da Saúde 25000.077928/2015-06Takeda Pharmaceuticals U.S.A. IISR-2020-103243University of South Florida (JEW) Robert A. Good Endowment
6 · The paper itself

Abstract

Severe combined immunodeficiency, SCID, is a pediatric emergency that represents the most critical group of inborn errors of immunity (IEI). Affected infants present with early onset life-threatening infections due to absent or non-functional T cells. Without early diagnosis and curative treatment, most die in early infancy. As most affected infants appear healthy at birth, newborn screening (NBS) is essential to identify and treat patients before the onset of symptoms. Here, we report 47 Brazilian patients investigated between 2009 and 2020 for SCID due to either a positive family history and/or clinical impression and low TRECs. Based on clinical presentation, laboratory finding, and genetic information, 24 patients were diagnosed as typical SCID, 14 as leaky SCID, and 6 as Omenn syndrome; 2 patients had non-SCID IEI, and 1 remained undefined. Disease onset median age was 2 months, but at the time of diagnosis and treatment, median ages were 6.5 and 11.5 months, respectively, revealing considerable delay which affected negatively treatment success. While overall survival was 51.1%, only 66.7% (30/45) lived long enough to undergo hematopoietic stem-cell transplantation, which was successful in 70% of cases. Forty-three of 47 (91.5%) patients underwent genetic testing, with a 65.1% success rate. Even though our patients did not come from the NBS programs, the diagnosis of SCID improved in Brazil during the pilot programs, likely due to improved medical education. However, we estimate that at least 80% of SCID cases are still missed. NBS-SCID started to be universally implemented in the city of São Paulo in May 2021, and it is our hope that other cities will follow, leading to early diagnosis and higher survival of SCID patients in Brazil.

Indexed as

Severe Combined ImmunodeficiencyBrazilChildDNAHumansInfantInfant, NewbornNeonatal ScreeningT-LymphocytesDNAInborn errors of immunityNewborn screeningPrimary immunodeficiencySCIDSevere combined immunodeficiencyTRECs

Identifiers

What OpenQuestion holds

Textmetadata
Read underepoch 390

Registered trials

None linked

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.