Evidence map›Paper›PMID 42871851›Full record

ArticleActa gastroenterologica Latinoamericana

[The Great Imposter: Three Cases of Inflammatory Bowel Disease Associated with Innate Immune Defects].

Diana Paola Sánchez Hernández, Sara Vallejo Tamayo

Abstract readCase ReportsEnglish Abstract
In one paragraph

Article in Acta gastroenterologica Latinoamericana. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

0numbers the graph read from it
0cells of the map it votes in
0citing papers in PubMed
–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

0 citing papers in PubMed.

No citing paper in PubMed yet.

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

2 authors.

Diana Paola Sánchez HernándezPediatra gastroenteróloga Hospital Sant Joan de Déu. Líder del programa Enfermedad Inflamatoria Intestinal en Pediatría, Hospital Pablo Tobón Uribe. Medellín, Colombia Hospital Pablo Tobón Uribe Medellín Colombia.ORCID https://orcid.org/0000-0002-4609-233X
Sara Vallejo TamayoResidente de Pediatría, Universidad CES. Medellín, Colombia Universidad CES Universidad CES Medellín Colombia.ORCID https://orcid.org/0000-0001-8548-9957

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Pediatric inflammatory bowel disease associated with inborn errors of immunity represents a diagnostic and therapeutic challenge, particularly in cases of early-onset, severe disease, extraintestinal manifestations, or refractoriness to treatment. We present three pediatric cases with an aggressive clinical course, multisystemic involvement, and an inadequate response to multiple therapeutic lines of therapy, in whom the persistent presence of warning signs prompted genetic testing. In one of these patients, exome sequencing identified a pathogenic variant in FOXP3, confirming the diagnosis of IPEX syndrome. In the other two patients, who were siblings, an alteration in XIAP was identified, consistent with a diagnosis of X-linked lymphoproliferative syndrome type 2. In the index patient with XIAP deficiency, the initial exome sequencing did not identify any causative variants. Subsequently, genome sequencing established the molecular diagnosis, which subsequently led to the cascade diagnosis of his sibling. These cases highlight the importance of early recognition of phenotypes suggestive of monogenic disease, maintaining diagnostic suspicion despite initially negative genetic test results, and conducting a timely multidisciplinary evaluation. Identification of an underlying inborn error of immunity can substantially modify the therapeutic approach, allow for the consideration of potentially curative options such as hematopoietic stem cell transplantation, and facilitate family genetic counseling.

Indexed as

inborn errors of immunityInflammatory bowel diseaseIPEX syndromemonogenic diseasepediatricsXIAP deficiency

Identifiers

PMID42871851
PMCPMC13660851

What OpenQuestion 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 OpenQuestion graph.