Evidence map›Paper›PMID 37739444›Full record

ArticleBMJ case reports2023

Congenital myopathy presenting as recurrent pneumonia with lung collapse and pulmonary artery hypertension.

Anand Vardhan, Devina Singh, Shalini Tripathi, Mala Kumar, Atin Singhai

Open access · greenAbstract readCase Reports
In one paragraph

Article in BMJ case reports, 2023. 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, top 61% 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

0 citing papers in PubMed, 0 citations in OpenAlex.

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

5 authors at 1 institution in 1 country.

Anand VardhanPediatrics, King George Medical College, Lucknow, Uttar Pradesh, India.ORCID http://orcid.org/0000-0001-7700-2336
Devina SinghPediatrics, King George Medical College, Lucknow, Uttar Pradesh, India singhdevina12@gmail.com shaliniawasthi@kgmcindia.edu mala_lko@yahoo.co.in.ORCID http://orcid.org/0000-0002-7122-7216
Shalini TripathiPediatrics, King George Medical College, Lucknow, Uttar Pradesh, India singhdevina12@gmail.com shaliniawasthi@kgmcindia.edu mala_lko@yahoo.co.in.ORCID http://orcid.org/0000-0003-0805-7370
Mala KumarPediatrics, King George Medical College, Lucknow, Uttar Pradesh, India singhdevina12@gmail.com shaliniawasthi@kgmcindia.edu mala_lko@yahoo.co.in.
Atin SinghaiPathology, King George Medical College, Lucknow, Uttar Pradesh, India.
King George's Medical University · IN

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

A boy presented with cough, breathlessness for 1 month, fever for 1 week with similar previous episodes without hospitalisation. He had generalised muscle wasting, acute chronic malnutrition and required immediate ventilation. Provisional diagnosis of recurrent pneumonia with failure to thrive was made. As serial chest X-rays showed recurrent lung collapse, congenital lung anomalies were ruled out. 2D-echocardiography showed pulmonary arterial hypertension. Workup for congenital immunodeficiency and cystic fibrosis was negative. There was no improvement in muscle mass despite total parenteral nutrition. He was noticed to have myopathic facies. History was reviewed when the mother reported reduced fetal movements in this pregnancy. The patient had low voice amplitude. Creatine kinase levels were normal. Muscle biopsy followed by whole exome sequencing identified frameshift duplication NM_020451.3(SELENON):c.249_250dupGG (p.Asp84Glyfs*17), thus, confirming diagnosis of SEPN1-related congenital myopathy (CM) with fibre-type disproportion. Respiratory system involvement was distracter, emphasising consideration of CM while evaluating persistent lung collapse with muscle wasting.

Indexed as

HypertensionMuscular DiseasesPneumoniaPulmonary Arterial HypertensionPulmonary AtelectasisFemaleHumansMaleMallory BodiesMuscular AtrophyMuscular DystrophiesPregnancyPulmonary ArteryScoliosisFailure to thriveNeuromuscular diseasePaediatrics (drugs and medicines)Pneumonia (infectious disease)Pulmonary hypertension

Identifiers

PMID37739444
PMCPMC10533661
OpenAlexW4386969802

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.