Evidence map›Paper›PMID 34310689›Full record

Trial reportClinical and experimental immunology2021

Clinical and laboratory findings in patients with leukocyte adhesion deficiency type I: A multicenter study in Turkey.

Ismail Yaz, Begum Ozbek, Hacer Neslihan Bildik, Cagman Tan, Sevil Oskay Halacli, Elif Soyak Aytekin, Saliha Esenboga, Sukru Cekic, Sara Sebnem Kilic, Ozlem Keskin and 4 more

Open access · bronzeAbstract readClinical TrialMulticenter Study
In one paragraph

Trial report in Clinical and experimental immunology, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 10 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
10citing papers in PubMed, 1 pooled it
1.4field-weighted citation impact, top 20% 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

10 citing papers in PubMed, 1 synthesis or guideline pooled it, 22 citations in OpenAlex.

  1. Pooled it
  2. Trial
  3. Article
  4. Article
  5. Article
  6. Article
  7. Article
  8. Article
  9. Rare and heterogeneous manifestations of leucocyte adhesion deficiency type 1: report of two cases with diagnostic dilemmas and novel ITGB2 mutation.Allergy, asthma, and clinical immunology : official journal of the Canadian Society of Allergy and Clinical Immunology · 2023
    Article
  10. 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

14 authors at 4 institutions in 2 countries.

Ismail YazDivision of Pediatric Immunology, Department of Pediatrics, Hacettepe University Medical School, Ankara, Turkey.ORCID 0000-0001-5278-7456
Begum OzbekDivision of Pediatric Immunology, Department of Pediatrics, Hacettepe University Medical School, Ankara, Turkey.ORCID 0000-0002-9053-2785
Hacer Neslihan BildikDivision of Pediatric Immunology, Department of Pediatrics, Hacettepe University Medical School, Ankara, Turkey.ORCID 0000-0002-2801-8494
Cagman TanDivision of Pediatric Immunology, Department of Pediatrics, Hacettepe University Medical School, Ankara, Turkey.ORCID 0000-0001-6972-1349
Sevil Oskay HalacliDivision of Pediatric Immunology, Department of Pediatrics, Hacettepe University Medical School, Ankara, Turkey.ORCID 0000-0003-1101-1779
Elif Soyak AytekinDivision of Pediatric Immunology, Department of Pediatrics, Hacettepe University Medical School, Ankara, Turkey.ORCID 0000-0002-2329-7543
Saliha EsenbogaDivision of Pediatric Immunology, Department of Pediatrics, Hacettepe University Medical School, Ankara, Turkey.ORCID 0000-0003-0562-9863
Sukru CekicDepartment of Pediatrics, Uludag University Medical School, Bursa, Turkey.ORCID 0000-0002-9574-1842
Sara Sebnem KilicDepartment of Pediatrics, Uludag University Medical School, Bursa, Turkey.ORCID 0000-0001-8571-2581
Ozlem KeskinDepartment of Pediatric Immunology and Allergy, Gaziantep University Medical School, Gaziantep, Turkey.ORCID 0000-0002-3061-7832
Karin van LeeuwenSanquin Research and Landsteiner Laboratory, Academic Medical Centre, University of Amsterdam, Amsterdam, the Netherlands.ORCID 0000-0001-7783-655X
Dirk RoosSanquin Research and Landsteiner Laboratory, Academic Medical Centre, University of Amsterdam, Amsterdam, the Netherlands.ORCID 0000-0001-5532-0202
Deniz CagdasDivision of Pediatric Immunology, Department of Pediatrics, Hacettepe University Medical School, Ankara, Turkey.ORCID 0000-0003-2213-4627
Ilhan TezcanDivision of Pediatric Immunology, Department of Pediatrics, Hacettepe University Medical School, Ankara, Turkey.ORCID 0000-0001-7257-4554
Hacettepe University · TRBursa Uludağ Üni̇versi̇tesi̇ · TRSanquin · NLGaziantep University · TR

Funding

Türkiye Bilimsel ve Teknolojik Araştirma Kurumu 315S125
6 · The paper itself

Abstract

Leukocyte adhesion deficiency type I is a rare primary immunodeficiency disorder characterized by mutations in the ITGB2 gene encoding CD18. We present clinical and immunological features of 15 patients with leukocyte adhesion deficiency type 1 (LAD-1). Targeted next-generation sequencing was performed with either a primary immunodeficiency gene panel comprising 266 genes or a small LAD-panel consisting of five genes for genetic analysis. To measure the expression level of integrins on the leukocyte surface, flow cytometry analysis was performed. The median age of the patients at diagnosis was 3 (1-48) months. Eleven (73%) of the 15 patients had a LAD-1 diagnosis in their first 6 months and 14 (93%) patients had consanguineous parents. Delayed separation of the umbilical cord was present in 80% (n = 12) of the patients in our cohort, whereas omphalitis was observed in 53% (n = 8) of the patients. Leukocytosis with neutrophil predominance was observed in 73% (n = 11) patients. Nine distinct variants in the ITGB2 gene in 13 of the 15 patients with LAD-1 were characterized, two of which (c.305_306delAA and c.779_786dup) are novel homozygous mutations of ITGB2. Four unrelated patients from Syria had a novel c.305_306delAA mutation that might be a founder effect for patients of Syrian origin. Four (27%) patients underwent hematopoietic stem cell transplantation. Two patients died because of HSCT complications and the other two are alive and well. Early differential diagnosis of the patients is critical in the management of the disease and genetic evaluation provides a basis for family studies and genetic counseling.

Indexed as

Genetic TestingHigh-Throughput Nucleotide SequencingLeukocyte-Adhesion Deficiency SyndromeMutationCD18 AntigensFemaleHumansInfantInfant, NewbornMaleTurkeyCD18 AntigensCD18HSCTintegrinITGB2leukocyte adhesion deficiency type 1 (LAD-1)

Identifiers

PMID34310689
PMCPMC8446394
OpenAlexW3138970460

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.