Evidence map›Paper›PMID 37238969›Full record

ArticleBiomedicines2023

Inborn Errors of Immunity in Children with Autoimmune and Allergic Complaints: A Single Center Experience from Diagnosis to Treatment.

Valentina Boz, Alessandra Tesser, Martina Girardelli, Francesca Burlo, Alessia Pin, Giovanni Maria Severini, Ginevra De Marchi, Federico Verzegnassi, Samuele Naviglio, Alberto Tommasini and 1 more

Open access · goldAbstract read
In one paragraph

Article in Biomedicines, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
0.2field-weighted citation impact, top 41% of its field
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

2 citing papers in PubMed, 1 citations in OpenAlex.

  1. Article
  2. 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

11 authors at 3 institutions in 1 country.

Valentina BozDepartment of Pediatrics, Institute for Maternal and Child Health IRCCS Burlo Garofolo, Via dell'Istria 65, 34137 Trieste, Italy.
Alessandra TesserDepartment of Pediatrics, Institute for Maternal and Child Health IRCCS Burlo Garofolo, Via dell'Istria 65, 34137 Trieste, Italy.ORCID 0000-0001-6267-3794
Martina GirardelliDepartment of Pediatrics, Institute for Maternal and Child Health IRCCS Burlo Garofolo, Via dell'Istria 65, 34137 Trieste, Italy.
Francesca BurloDepartment of Medical, Surgical and Health Sciences, University of Trieste, 34137 Trieste, Italy.ORCID 0000-0002-8915-5855
Alessia PinDepartment of Pediatrics, Institute for Maternal and Child Health IRCCS Burlo Garofolo, Via dell'Istria 65, 34137 Trieste, Italy.ORCID 0000-0002-0145-1976
Giovanni Maria SeveriniDepartment of Pediatrics, Institute for Maternal and Child Health IRCCS Burlo Garofolo, Via dell'Istria 65, 34137 Trieste, Italy.ORCID 0000-0002-5790-2000
Ginevra De MarchiRheumatology Clinic, Department of Medical and Biological Sciences, Azienda Sanitaria Universitaria Friuli Centrale c/o, University of Udine, 33100 Udine, Italy.ORCID 0000-0002-3087-8477
Federico VerzegnassiDepartment of Pediatrics, Institute for Maternal and Child Health IRCCS Burlo Garofolo, Via dell'Istria 65, 34137 Trieste, Italy.
Samuele NaviglioDepartment of Pediatrics, Institute for Maternal and Child Health IRCCS Burlo Garofolo, Via dell'Istria 65, 34137 Trieste, Italy.ORCID 0000-0002-1915-9965
Alberto TommasiniDepartment of Pediatrics, Institute for Maternal and Child Health IRCCS Burlo Garofolo, Via dell'Istria 65, 34137 Trieste, Italy.ORCID 0000-0002-6943-7927
Erica ValencicDepartment of Pediatrics, Institute for Maternal and Child Health IRCCS Burlo Garofolo, Via dell'Istria 65, 34137 Trieste, Italy.ORCID 0000-0002-7380-8014
IRCCS Materno Infantile Burlo Garofolo · ITUniversity of Trieste · ITUniversity of Udine · IT

Funding

IRCCS Materno Infantile Burlo Garofolo RC 24/17Ministero della Salute RF-2016-02362384
6 · The paper itself

Abstract

Inborn errors of immunity (IEI) associated with immune dysregulation are not sufficiently addressed in shared recommendation, resulting in delayed diagnosis and high morbidity. The availability of precision medicine for some of these immune defects makes it urgent to evaluate effective strategies to diagnose and treat such defects before the occurrence of severe complications. A diagnosis of an IEI in these patients enabled the use of a more specific treatment in most cases, and these have the potential to prevent further disease progression. We studied immune dysregulation diseases in 30 patients with autoimmune or allergic phenotypes, exploiting data from clinics and immunophenotype, genetic and transcriptome investigations, and 6 of them were diagnosed with a monogenic disorder. Our results confirm that a non-negligible number of children with IEIs may present with signs and symptoms of immune dysregulation and share many features with common multifactorial immune conditions. Reaching a genetic diagnosis becomes more likely in the presence of multiple clinical manifestations, especially when in association with abnormalities of lymphocytes subsets and/or immunoglobulins levels. Moreover, 5 of 6 patients that obtained a diagnosis of monogenic disorder received precision therapy, in four cases with a good or moderate response.

Indexed as

allergyautoimmunityimmunodysregulationinborn errors of immunityprecision treatmentsprimary immune deficiency

Identifiers

PMID37238969
PMCPMC10215820
OpenAlexW4367316223

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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 OpenQuestion graph.