Evidence map›Paper›PMID 28272068›Full record

ArticleJournal of postgraduate medicine

Rare disease heralded by pulmonary manifestations: Avoiding pitfalls of an "asthma" label.

S Bajaj, M Muranjan, S Karande, D Prabhat

Open access · diamondAbstract readCase Reports
In one paragraph

Article in Journal of postgraduate medicine. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 4 papers.

0numbers the graph read from it
0cells of the map it votes in
4citing papers in PubMed
0.8field-weighted citation impact, top 29% 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

4 citing papers in PubMed, 11 citations in OpenAlex.

  1. Review
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4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

4 authors.

S BajajDepartment of Pediatrics, Genetic Clinic, Seth G.S. Medical College and KEM Hospital, Mumbai, Maharashtra, India.
M MuranjanDepartment of Pediatrics, Genetic Clinic, Seth G.S. Medical College and KEM Hospital, Mumbai, Maharashtra, India.
S KarandeDepartment of Pediatrics, Genetic Clinic, Seth G.S. Medical College and KEM Hospital, Mumbai, Maharashtra, India.
D PrabhatDepartment of Pathology, Seth G.S. Medical College and KEM Hospital, Mumbai, Maharashtra, India.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Pulmonary manifestations are seldom recognized as symptoms of storage disorders. The report describes the diagnostic journey in a 30-month-old male infant, born of a third-degree consanguineous marriage referred to our institute as severe persistent asthma. History revealed that the child had progressively worsening breathlessness and persistent dry cough not associated with fever but accompanied by weight loss. On physical examination, there was growth failure, respiratory distress, clubbing, hepatosplenomegaly, and occasional rhonchi. Blood gas revealed hypoxemia which improved with oxygen administration. Plain X-rays and high-resolution computed tomography of the chest showed perihilar alveolar infiltrates and patchy consolidation. The clinicoradiological features did not support a diagnosis of asthma but favored interstitial lung disease (ILD). Bronchoalveolar lavage was performed as a first-tier investigation. It showed periodic acid-Schiff-negative foamy macrophages. The clues of consanguinity, visceromegaly, ILD, and foamy macrophages in the bronchoalveolar fluid prompted consideration of lysosomal storage disorders as the likely etiology. Gaucher disease and Niemann-Pick disease A/B were ruled out by enzyme estimation. Niemann-Pick disease type C was suspected and confirmed by detecting a homozygous mutation in the NPC2 gene. This case serves to caution physicians against labeling breathlessness in every toddler as asthma. It emphasizes the importance of searching for tell-tale signs such as clubbing and extrapulmonary clues which point to a systemic disease such as lysosomal storage disorders as a primary etiology of chronic respiratory symptoms.

Indexed as

AsthmaBronchoalveolar LavageCarrier ProteinsChild, PreschoolConsanguinityCoughDyspneaGlycoproteinsHumansLung DiseasesMaleNiemann-Pick Disease, Type CRare DiseasesRespiratory Tract InfectionsVesicular Transport ProteinsCarrier ProteinsGlycoproteinsNPC2 protein, humanVesicular Transport Proteins

Identifiers

PMID28272068
PMCPMC5414422
OpenAlexW2597171179

What OpenQuestion holds

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LicenceCC BY-NC-SA
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.