Evidence map›Paper›PMID 27064303›Full record

ArticleUpsala journal of medical sciences2016

The appearance of newly identified intraocular lesions in Gaucher disease type 3 despite long-term glucocerebrosidase replacement therapy.

Nadia Sawicka-Gutaj, Maciej Machaczka, Izabela Kulińska-Niedziela, Jadwiga Bernardczyk-Meller, Paweł Gutaj, Jerzy Sowiński, Marek Ruchała

Open access · diamondAbstract readCase Reports
In one paragraph

Article in Upsala journal of medical sciences, 2016. 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
1.4field-weighted citation impact, top 19% 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, 13 citations in OpenAlex.

  1. Review
  2. Review
  3. Article
  4. Photo Essay: Retinal Changes in Type 3 Gaucher Disease.Neuro-ophthalmology (Aeolus Press) · 2018
    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

7 authors at 3 institutions in 2 countries.

Nadia Sawicka-Gutaja Department of Endocrinology, Metabolism and Internal Medicine , Poznan University of Medical Sciences , Poznan , Poland ;
Maciej Machaczkab Hematology Center Karolinska and Department of Medicine at Huddinge , Karolinska Institutet, Karolinska University Hospital Huddinge , Stockholm , Sweden ;
Izabela Kulińska-Niedzielac Heliodor Swiecicki Clinical Hospital, Ophthalmology Outpatient Clinic, Poznan University of Medical Sciences , Poznan , Poland ;
Jadwiga Bernardczyk-Mellerd Department of Ophthalmology , NZOZ 'OCU SERVICE' , Poznan , Poland ;
Paweł Gutaje Department of Obstetrics and Women's Diseases , Poznan University of Medical Sciences , Poznan , Poland.
Jerzy Sowińskia Department of Endocrinology, Metabolism and Internal Medicine , Poznan University of Medical Sciences , Poznan , Poland ;
Marek Ruchałaa Department of Endocrinology, Metabolism and Internal Medicine , Poznan University of Medical Sciences , Poznan , Poland ;
Poznan University of Medical Sciences · PLHeliodor Swiecicki Clinical Hospital · PLKarolinska Institutet · SE

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Background Gaucher disease (GD) is an autosomal recessive lipid storage disorder caused by the deficient activity of the lysosomal enzyme glucocerebrosidase. The presence of central nervous system disease is a hallmark of the neuronopathic forms of GD (types 2 and 3). Intraocular lesions (e.g. corneal clouding, retinal lesions, and vitreous opacities) have been infrequently reported in GD type 3 (GD3). Moreover, there are virtually no published data on the occurrence and natural course of intraocular lesions in GD3 patients treated with enzyme replacement therapy (ERT). Case presentation We describe the case of a 26-year-old Polish male with L444P homozygous GD3 (mutation c.1448T > C in the GBA1 gene) who developed fundus lesions despite 10 years of ERT. At the age of 23 years, a spectral domain optical coherence tomography (OCT) examination was performed which disclosed the presence of discrete lesions located preretinally, intraretinally in the nerve fiber layer, and in the vitreous body. A 3-year follow-up OCT examination has not shown any significant progression of the fundus lesions. Conclusions To the best of our knowledge, this is the first published report describing the occurrence of newly identified retinal and preretinal lesions occurring during long-term ERT in GD3. We recommend that a careful ophthalmic assessment, including a dilated fundus examination, should be included as part of annual follow-up in patients with GD3. Further studies are needed to understand the nature and clinical course of these changes and whether or not these intraocular findings have any predictive value in the context of neurologic and skeletal progression in GD3.

Indexed as

Enzyme Replacement TherapyAdultGaucher DiseaseGlucosylceramidaseHomozygoteHumansMalePolandRetinal DiseasesTomography, Optical CoherenceGlucosylceramidaseEnzyme replacement therapyGaucher disease type 3intraocular lesionsneuronopathicoptical coherence tomographyretina

Identifiers

PMID27064303
PMCPMC4967266
OpenAlexW2322964909

What OpenQuestion holds

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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.