Evidence map›Paper›PMID 36779112›Full record

ArticleCureus2023

Niemann-Pick Disease: A Case Report and Literature Review.

Paola Jacqueline Vélez Pinos, Michell Susan Saavedra Palacios, Paolo Andrés Colina Arteaga, Tania Diciana Arevalo Cordova

Abstract readCase Reports
In one paragraph

Article in Cureus, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 7 papers.

0numbers the graph read from it
0cells of the map it votes in
7citing 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

7 citing papers in PubMed.

  1. Article
  2. Review
  3. Article
  4. Article
  5. Review
  6. Article
  7. Article
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.

Paola Jacqueline Vélez PinosPediatrics, University of Azuay, Cuenca, ECU.
Michell Susan Saavedra PalaciosCollege of Medicine, University of Cuenca, Cuenca, ECU.
Paolo Andrés Colina ArteagaCollege of Medicine, Catholic University of Santiago de Guayaquil, Guayaquil, ECU.
Tania Diciana Arevalo CordovaMedicine, Technical University of Machala, Machala, ECU.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Niemann-Pick disease (NPD) A/B is a lysosomal storage disease (LSD), caused by an autosomal recessive disorder that causes variation in sphingomyelin phosphodiesterase-1 (SMPD1). Systemic signs are cholestatic jaundice in the neonatal period or hepatosplenomegaly in infancy. The clinical course experienced by our patient did not correspond to the classic phenotypes. The diagnosis was effectively made at four years and three months of age when different signs such as abdominal distension, hepatosplenomegaly, and chronic malnutrition were present. Given the high suspicion of metabolic storage disease, an enzyme activity study, liver and bone marrow biopsies, and molecular studies were performed. In the bone marrow biopsy, pseudo-Gaucher foam cells were observed. Additionally, the liver biopsy showed dispersed ballooned cells with deposit material and nested cells with granular material. The double enzymatic assay was ordered to determine if the cause of these findings was due to Niemann-Pick or Gaucher disease; decreased sphingomyelinase activity values were obtained (0.28 mcoml/L/h). Subsequently, the molecular genetics study reported a double alteration in the sequence that encodes the SMPD1 gene, located on chromosome 11p15.4, which confirmed NPD type A or B. The overlap and the lack of some findings made the diagnosis very difficult. Diagnosis is crucial due to the multisystem involvement that this LSD can have.

Indexed as

inborn errorslysosomal storage diseasesmetabolismniemann-pick diseasesphingolipidoses

Identifiers

PMID36779112
PMCPMC9906968

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