Evidence map›Paper›PMID 39119442›Full record

ArticleCureus2024

Cases of Fabry Disease in Which Pathogenic Variants Are Not Detected in Parent-Child Pairs.

Naoki Akeho, Kumiko Muta, Kenta Torigoe, Mineaki Kitamura, Takaaki Sawada, Kimitoshi Nakamura, Hiroshi Mukae, Tomoya Nishino

Abstract readCase Reports
In one paragraph

Article in Cureus, 2024. 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
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.

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

8 authors.

Naoki AkehoDepartment of Nephrology, Izumikawa Hospital, Minamishimabara, JPN.
Kumiko MutaDepartment of Nephrology, Nagasaki University Hospital, Nagasaki, JPN.
Kenta TorigoeDepartment of Nephrology, Nagasaki University Hospital, Nagasaki, JPN.
Mineaki KitamuraDepartment of Nephrology, Nagasaki University Hospital, Nagasaki, JPN.
Takaaki SawadaDepartment of Pediatrics, Faculty of Life Sciences, Kumamoto University, Kumamoto, JPN.
Kimitoshi NakamuraDepartment of Pediatrics, Faculty of Life Sciences, Kumamoto University, Kumamoto, JPN.
Hiroshi MukaeDepartment of Respiratory Medicine, Nagasaki University Graduate School of Biomedical Sciences, Nagasaki, JPN.
Tomoya NishinoDepartment of Nephrology, Nagasaki University Hospital, Nagasaki, JPN.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

A 15-year-old male has been experiencing fever, limb pain during exercise, and reduced sweating since childhood. During an investigation into his fever, a family history of Fabry disease was discovered, prompting a referral to our department. He was diagnosed with Fabry disease based on decreased alpha-galactosidase A (α-Gal A) activity. Concurrently, his mother was found to have experienced limb pain during fevers since childhood, and she was also diagnosed with Fabry disease based on decreased α-Gal A activity. In the genetic analysis of both individuals, the IVS1+17A>G GLA variant was identified. This variant is considered benign and not classified as a pathogenic variant. Enzyme replacement therapy has been effective in improving clinical symptoms. His sister, who has not been diagnosed with Fabry disease due to normal clinical symptoms and α-GAL A activity, also had the same variant. Among the various GLA variants, many are classified as benign rather than pathogenic. In the present cases, the possibility of other factors that cannot be identified by genetic analysis is suggested, making this case significant and worth reporting.

Indexed as

enzyme replacement therapyfabry diseasegla variantlyso-gb3α-galactosidase activity

Identifiers

PMID39119442
PMCPMC11307133

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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.