Evidence map›Paper›PMID 34502390›Full record

ArticleInternational journal of molecular sciences2021

Atypical Hemolytic Uremic Syndrome (aHUS) and Adenosine Deaminase (ADA)-Deficient Severe Combined Immunodeficiency (SCID)-Two Diseases That Exacerbate Each Other: Case Report.

Anna Bogdał, Andrzej Badeński, Małgorzata Pac, Anna Wójcicka, Marta Badeńska, Agnieszka Didyk, Elżbieta Trembecka-Dubel, Nel Dąbrowska-Leonik, Małgorzata Walaszczyk, Natalia Matysiak and 3 more

Open access · goldAbstract readCase Reports
In one paragraph

Article in International journal of molecular sciences, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.

0numbers the graph read from it
0cells of the map it votes in
3citing papers in PubMed
0.2field-weighted citation impact, top 46% 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

3 citing papers in PubMed, 4 citations in OpenAlex.

  1. Review
  2. Review
  3. 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

13 authors at 2 institutions in 1 country.

Anna BogdałDistrict Hospital in Zawiercie, ul. Miodowa 14, 42-400 Zawiercie, Poland.
Andrzej BadeńskiDepartment of Pediatrics, Faculty of Medical Sciences in Zabrze, Medical University of Silesia in Katowice, ul. 3 Maja 13/15, 41-800 Zabrze, Poland.ORCID 0000-0001-6947-005X
Małgorzata PacDepartment of Immunology, The Children's Memorial Health Institute, 04-730 Warsaw, Poland.ORCID 0000-0003-2071-3297
Anna WójcickaWarsaw Genomics, 01-682 Warsaw, Poland.
Marta BadeńskaDepartment of Pediatrics, Faculty of Medical Sciences in Zabrze, Medical University of Silesia in Katowice, ul. 3 Maja 13/15, 41-800 Zabrze, Poland.ORCID 0000-0003-1711-8067
Agnieszka DidykDepartment of Pediatric Nephrology with Dialysis Division for Children, Public Clinical Hospital No. 1 in Zabrze, 41-800 Zabrze, Poland.
Elżbieta Trembecka-DubelDepartment of Pediatric Nephrology with Dialysis Division for Children, Public Clinical Hospital No. 1 in Zabrze, 41-800 Zabrze, Poland.
Nel Dąbrowska-LeonikDepartment of Immunology, The Children's Memorial Health Institute, 04-730 Warsaw, Poland.
Małgorzata WalaszczykDepartment of Anaesthesiology and Intensive Therapy, Faculty of Medical Sciences in Zabrze, Medical University of Silesia in Katowice, ul. 3 Maja 13/15, 41-800 Zabrze, Poland.
Natalia MatysiakDepartment of Histology and Cell Pathology, Faculty of Medical Sciences in Zabrze, Medical University of Silesia in Katowice, ul. 3 Maja 13/15, 41-800 Zabrze, Poland.
Aurelia Morawiec-KnysakDepartment of Pediatric Nephrology with Dialysis Division for Children, Public Clinical Hospital No. 1 in Zabrze, 41-800 Zabrze, Poland.
Tomasz SzczepańskiDepartment of Pediatric Hematology and Oncology, Faculty of Medical Sciences in Zabrze, Medical University of Silesia in Katowice ul. 3 Maja 13/15, 41-800 Zabrze, Poland.ORCID 0000-0001-5336-261X
Maria SzczepańskaDepartment of Pediatrics, Faculty of Medical Sciences in Zabrze, Medical University of Silesia in Katowice, ul. 3 Maja 13/15, 41-800 Zabrze, Poland.ORCID 0000-0002-6772-1983
Medical University of Silesia · PLChildren's Memorial Health Institute · PL

Funding

POIR.04.04.00-00-41DB/17-00 , TEAM TECH 4/2017 for Anna Wójcicka
6 · The paper itself

Abstract

Hemolytic uremic syndrome (HUS) is defined by the triad of microangiopathic hemolytic anemia, thrombocytopenia, and acute kidney injury (AKI). Atypical HUS (aHUS), distinguished by its etiology, is caused by uncontrolled overactivation of the alternative complement pathway. The correct diagnosis of aHUS is complex and involves various gene mutations. Severe combined immunodeficiency (SCID), characterized by severe T-cell lymphocytopenia and a lack of antigen-specific T-cell and B-cell immune responses, is of seldom occurrence. In 10-15% of pediatric patients, SCID is caused by adenosine deaminase (ADA) deficiency. The authors describe the case of a boy who suffered from both aHUS and ADA-deficient SCID. At the age of 9 months, the patient presented acute kidney injury with anuria and coagulopathy. The diagnosis of aHUS was established on the basis of alternative complement pathway deregulation and disease-associated gene mutations. Further examination revealed immune system failure and, at the age of 13 months, the ADA deficiency was confirmed by genetic tests and the boy was diagnosed with ADA-SCID. ADA SCID has recently been described as a possible triggering factor of aHUS development and progression. However, more research is required in this field. Nevertheless, it is crucial in clinical practice to be aware of these two co-existing life-threatening diseases.

Indexed as

Acute Kidney InjuryAdenosine DeaminaseAgammaglobulinemiaAnemia, HemolyticAtypical Hemolytic Uremic SyndromeComorbidityHumansInfantMaleMutationSevere Combined ImmunodeficiencyThrombocytopeniaThrombotic MicroangiopathiesAdenosine Deaminaseadenosine deaminase deficiencyatypical hemolytic uremic syndromechildrensevere combined immunodeficiencythrombotic microangiopathy

Identifiers

PMID34502390
PMCPMC8430959
OpenAlexW3198565465

What OpenQuestion holds

Textmetadata
LicenceCC BY
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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.