Evidence map›Paper›PMID 41096571›Full record

ArticleInternational journal of molecular sciences2025

13q Deletion Syndrome Presenting with Lymphopenia Detected Through Newborn Screening for Primary Immunodeficiencies.

Irina Efimova, Anna Mukhina, Zhanna Markova, Sergey Mordanov, Irina Soprunova, Dmitry Pershin, Natalya Balinova, Yunna Petrusenko, Dmitry Meleshko, Rena Zinchenko and 5 more

Abstract readCase Reports
In one paragraph

Article in International journal of molecular sciences, 2025. 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
–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

2 citing papers in PubMed.

  1. Article
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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

15 authors.

Irina EfimovaResearch Centre for Medical Genetics, Moscow 115522, Russia.ORCID 0000-0003-4672-1447
Anna MukhinaResearch Centre for Medical Genetics, Moscow 115522, Russia.
Zhanna MarkovaResearch Centre for Medical Genetics, Moscow 115522, Russia.ORCID 0000-0003-2941-2861
Sergey MordanovMedical Genetics Laboratory, Rostov State Medical University, Rostov-on-Don 344022, Russia.
Irina SoprunovaCenter for Family Health and Reproduction, Astrakhan 414040, Russia.
Dmitry PershinDmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology, and Immunology, Moscow 117198, Russia.ORCID 0000-0002-6148-7209
Natalya BalinovaResearch Centre for Medical Genetics, Moscow 115522, Russia.ORCID 0000-0001-9493-6544
Yunna Petrusenko"Biotech Campus" LLC, Moscow 117437, Russia.ORCID 0009-0002-8318-5241
Dmitry Meleshko"Biotech Campus" LLC, Moscow 117437, Russia.
Rena ZinchenkoResearch Centre for Medical Genetics, Moscow 115522, Russia.ORCID 0000-0003-3586-3458
Nadezhda ShilovaResearch Centre for Medical Genetics, Moscow 115522, Russia.ORCID 0000-0002-0641-1084
Sergey VoroninResearch Centre for Medical Genetics, Moscow 115522, Russia.
Anna ShcherbinaDmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology, and Immunology, Moscow 117198, Russia.
Sergey KutsevResearch Centre for Medical Genetics, Moscow 115522, Russia.
Andrey MarakhonovResearch Centre for Medical Genetics, Moscow 115522, Russia.ORCID 0000-0002-0972-5118

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

The expanded newborn screening (NBS) program in the Russian Federation, launched in 2023, includes the detection of severe forms of T- and B-cell immunodeficiencies via TREC/KREC quantification. We report a rare case of a male infant having multiple congenital anomalies and lymphopenia identified through this program. Genetic testing revealed a 25.8 Mb terminal deletion spanning 13q31.2-qter, consistent with 13q deletion syndrome. Initial NBS revealed reduced TREC levels, prompting further evaluation. The patient exhibited a complex phenotype, including central nervous system malformation (alobar holoprosencephaly), severe congenital heart disease, renal hypoplasia, limb and genitourinary anomalies, and facial dysmorphism. Postnatal complications included pneumonia, pleuritis, and chylothorax. Flow cytometry demonstrated mild T- and B-cell lymphopenia. The genomic defect was characterized using long-read third-generation sequencing, enabling precise breakpoint identification and accurate mapping of deleted genes. The deletion was confirmed via subtelomeric FISH analysis. The patient died at 7 months of age due to the progression of underlying congenital anomalies and associated complications. Our findings broaden the clinical characterization of distal 13q deletion syndrome and demonstrate the value of long-read sequencing in structural chromosomal analysis. They further highlight the difficulties of caring for neonates having complex malformations and immune dysfunction. Given the potential for both primary and secondary immune disturbances, comprehensive immunological evaluation should be considered in patients having 13q deletion syndrome to improve diagnostic accuracy and inform appropriate clinical management.

Indexed as

Chromosome DisordersLymphopeniaPrimary Immunodeficiency DiseasesChromosome DeletionChromosomes, Human, Pair 13HumansInfantInfant, NewbornMaleNeonatal Screening13q deletion syndromelong-read sequencingnewborn screeningprimary immunodeficiencyTREC/KREC assay

Identifiers

PMID41096571
PMCPMC12524871

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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.