Evidence map›Paper›PMID 38929247›Full record

ReviewChildren (Basel, Switzerland)2024

Lung Diseases and Rare Disorders: Is It a Lysosomal Storage Disease? Differential Diagnosis, Pathogenetic Mechanisms and Management.

Chiara Montanari, Veronica Maria Tagi, Enza D'Auria, Vincenzo Guaia, Anna Di Gallo, Michele Ghezzi, Elvira Verduci, Laura Fiori, Gianvincenzo Zuccotti

Abstract readReview
In one paragraph

Review in Children (Basel, Switzerland), 2024. 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
–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

4 citing papers in PubMed.

  1. Article
  2. Review
  3. Article
  4. Review
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

9 authors.

Chiara MontanariDepartment of Pediatrics, Vittore Buzzi Children's Hospital, University of Milan, 20154 Milan, Italy.ORCID 0000-0002-4875-6179
Veronica Maria TagiDepartment of Pediatrics, Vittore Buzzi Children's Hospital, University of Milan, 20154 Milan, Italy.ORCID 0000-0002-7001-9191
Enza D'AuriaDepartment of Pediatrics, Vittore Buzzi Children's Hospital, University of Milan, 20154 Milan, Italy.ORCID 0000-0003-2750-5810
Vincenzo GuaiaDepartment of Pediatrics, Vittore Buzzi Children's Hospital, University of Milan, 20154 Milan, Italy.ORCID 0009-0004-0710-3413
Anna Di GalloDepartment of Pediatrics, Vittore Buzzi Children's Hospital, University of Milan, 20154 Milan, Italy.
Michele GhezziDepartment of Pediatrics, Vittore Buzzi Children's Hospital, University of Milan, 20154 Milan, Italy.ORCID 0000-0002-7434-9112
Elvira VerduciDepartment of Health Sciences, University of Milan, 20146 Milan, Italy.ORCID 0000-0003-2111-3111
Laura FioriDepartment of Pediatrics, Vittore Buzzi Children's Hospital, University of Milan, 20154 Milan, Italy.ORCID 0000-0003-3579-7403
Gianvincenzo ZuccottiDepartment of Pediatrics, Vittore Buzzi Children's Hospital, University of Milan, 20154 Milan, Italy.ORCID 0000-0002-2795-9874

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Pulmonologists may be involved in managing pulmonary diseases in children with complex clinical pictures without a diagnosis. Moreover, they are routinely involved in the multidisciplinary care of children with rare diseases, at baseline and during follow-up, for lung function monitoring. Lysosomal storage diseases (LSDs) are a group of genetic diseases characterised by a specific lysosomal enzyme deficiency. Despite varying pathogen and organ involvement, they are linked by the pathological accumulation of exceeding substrates, leading to cellular toxicity and subsequent organ damage. Less severe forms of LSDs can manifest during childhood or later in life, sometimes being underdiagnosed. Respiratory impairment may stem from different pathogenetic mechanisms, depending on substrate storage in bones, with skeletal deformity and restrictive pattern, in bronchi, with obstructive pattern, in lung interstitium, with altered alveolar gas exchange, and in muscles, with hypotonia. This narrative review aims to outline different pulmonary clinical findings and a diagnostic approach based on key elements for differential diagnosis in some treatable LSDs like Gaucher disease, Acid Sphingomyelinase deficiency, Pompe disease and Mucopolysaccharidosis. Alongside their respiratory clinical aspects, which might overlap, we will describe radiological findings, lung functional patterns and associated symptoms to guide pediatric pulmonologists in differential diagnosis. The second part of the paper will address follow-up and management specifics. Recent evidence suggests that new therapeutic strategies play a substantial role in preventing lung involvement in early-treated patients and enhancing lung function and radiological signs in others. Timely diagnosis, driven by clinical suspicion and diagnostic workup, can help in treating LSDs effectively.

Indexed as

Acid sphingomyelinase deficiencydiagnosisGaucher diseaselungLysosomal storage diseasesmucopolysaccharidosisPompe diseaserespiratory systemtreatment

Identifiers

PMID38929247
PMCPMC11201433

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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.