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ArticleBiomedica : revista del Instituto Nacional de Salud2024

First report of PURA syndrome in a Colombian patient with de novo missense variant c.692T>C (p.Phe231Ser)

Sandra Milena Cerón, Daniel Alejandro Pérez, Julio Herberth Montaño, María Amparo Acosta

Abstract readCase ReportsEnglish Abstract
In one paragraph

Article in Biomedica : revista del Instituto Nacional de Salud, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed, 1 pooled it
–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

1 citing paper in PubMed, 1 synthesis or guideline pooled it.

  1. Pooled it
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

4 authors.

Sandra Milena CerónFacultad de Ciencias de la Salud, Universidad del Cauca, Popayán, Colombia.ORCID 0009-0004-0055-7164
Daniel Alejandro PérezFacultad de Ciencias de la Salud, Universidad del Cauca, Popayán, Colombia.ORCID 0000-0002-6646-441X
Julio Herberth MontañoFacultad de Ciencias de la Salud, Universidad del Cauca, Popayán, Colombia.ORCID 0009-0009-0766-0662
María Amparo AcostaDepartamento de Pediatría, Facultad de Ciencias de la Salud, Universidad del Cauca, Popayán, Colombia; Hospital Universitario San José, Popayán, Cauca, Colombia.ORCID 0000-0002-0513-8127

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

We present the first documented case of PURA syndrome in Colombia. This rare neurological disease results from mutations in the PURA gene located on chromosome 5, leading to haploinsufficiency of the PUR-α protein. This protein is essential for early brain development and neuronal function. The patient, a seven-years-old boy, started showing dystonic hand movements at 14 days of age; at six, he had neurodevelopmental delay, generalized hypotonia, frequent episodes of apnea, and swallowing difficulties. Although other conditions were initially considered, such as Duchenne muscular dystrophy and neuronal ceroid lipofuscinosis, a whole exome sequencing revealed the pathogenic variant c.692T>C (p.Phe231Ser) in the exon 1 of the PURA gene, not previously reported in other patients. With this finding, we adopted a comprehensive management approach addressing the patient’s characteristics and alterations. Since the PURA syndrome is not on the list of orphan/rare diseases recognized by the Colombian Ministerio de Salud y Protección Social, we hope our report will contribute to its official recognition. The case shows the importance of considering rare diagnoses in patients with uncommon neurological symptoms, underlining the usefulness of genomic sequencing in diagnosis and the need for collaboration to optimize healthcare for patients with PURA syndrome and similar diseases.

Indexed as

HaploinsufficiencyAmino Acid SubstitutionChildColombiaFemaleHumansMaleMutation, MissenseChildNervous system diseasesneurodevelopmental disorders

Identifiers

PMID39531550
PMCPMC11781598

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