Evidence map›Paper›PMID 39639321›Full record

ArticleMolecular cytogenetics2024

Clinical and genomic profiling of a patient with a de novo ring chromosome 18: a case report highlighting autoimmune and neurological implications.

Annalaura Montanari, Paola Caforio, Annalisa Paparella, Paola Casieri, Maria Cristina Nuzzi, Maria Fatima Antonucci, Claudia Rita Catacchio, Marilina Tampoia, Mattia Gentile, Roberta Bucci and 3 more

Abstract read
In one paragraph

Article in Molecular cytogenetics, 2024. 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

13 authors.

Annalaura Montanari *Dipartimento di Bioscienze, Biotecnologie e Ambiente, Università degli Studi di Bari "Aldo Moro", Bari, Italy.
Paola Caforio *U.O.C Patologia Clinica - Sezione di Genetica Medica, Ospedale SS. Annunziata Taranto - ASL Taranto, Taranto, Italy.
Annalisa PaparellaDipartimento di Bioscienze, Biotecnologie e Ambiente, Università degli Studi di Bari "Aldo Moro", Bari, Italy.
Paola CasieriU.O.C Patologia Clinica - Sezione di Genetica Medica, Ospedale SS. Annunziata Taranto - ASL Taranto, Taranto, Italy.
Maria Cristina NuzziU.O.C Patologia Clinica - Sezione di Genetica Medica, Ospedale SS. Annunziata Taranto - ASL Taranto, Taranto, Italy.
Maria Fatima AntonucciU.O.C Patologia Clinica - Sezione di Genetica Medica, Ospedale SS. Annunziata Taranto - ASL Taranto, Taranto, Italy.
Claudia Rita CatacchioDipartimento di Bioscienze, Biotecnologie e Ambiente, Università degli Studi di Bari "Aldo Moro", Bari, Italy.
Marilina TampoiaU.O.C Patologia Clinica - Sezione di Genetica Medica, Ospedale SS. Annunziata Taranto - ASL Taranto, Taranto, Italy.
Mattia GentileU.O.C. Laboratorio di Genetica Medica, PO Di Venere - ASL Bari, Bari, Italy.
Roberta BucciU.O.C. Laboratorio di Genetica Medica, PO Di Venere - ASL Bari, Bari, Italy.
Valerio CecinatiU.O.C Patologia Clinica - Sezione di Genetica Medica, Ospedale SS. Annunziata Taranto - ASL Taranto, Taranto, Italy.
Angelo CellamareU.O.C Patologia Clinica - Sezione di Genetica Medica, Ospedale SS. Annunziata Taranto - ASL Taranto, Taranto, Italy. angelo.cellamare@asl.taranto.it.
Francesca AntonacciDipartimento di Bioscienze, Biotecnologie e Ambiente, Università degli Studi di Bari "Aldo Moro", Bari, Italy. francesca.antonacci@uniba.it.

Funding

Ministero dell'Università e della Ricerca CUP H53D23007700001
6 · The paper itself

Abstract

Ring chromosome 18 (r(18)) is a rare chromosomal abnormality characterized by the circular rearrangement of chromosome 18, which presents significant challenges in genotype-phenotype correlations due to variability in deletions across the 18p and 18q arms. We report the case of a pediatric patient with a de novo ring chromosome 18, diagnosed by karyotype analysis and confirmed by high-resolution SNP arrays. The patient exhibited pathogenic copy number variants (CNVs) in the 18p11.32p11.22 and 18q23 regions, involving 36 and 10 OMIM genes, respectively. Clinically, the patient presented with hypothyroidism secondary to autoimmune thyroiditis, autoimmune hepatitis type II, and genetic predisposition to celiac disease and insulin-dependent diabetes mellitus (IDDM) along with notable dysmorphic features. The 18q microdeletion encompasses the MBP gene, involved in the development and functionality of the nervous system, as supported by hypotonia and gliosis shown by the MRI. This case highlights the complex interplay between genetic imbalances on chromosome 18 and autoimmune phenotypes, emphasizing the need for ongoing research to elucidate underlying mechanisms and optimize clinical management for individuals with r(18).

Identifiers

PMID39639321
PMCPMC11619688

What OpenQuestion holds

Textmetadata
LicenceCC BY
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.