Evidence map›Paper›PMID 38952472›Full record

ReviewFrontiers in neurology2024

Case report: Cerebrotendinous xanthomatosis treatment follow-up.

Karolina Ejsmont-Sowała, Tomasz Książek, Katarzyna Maciorowska-Rosłan, Joanna Rosłan, Agata Czarnowska, Anna Jakubiuk-Tomaszuk, Joanna Tarasiuk, Katarzyna Kapica-Topczewska, Alina Kułakowska

Abstract readReview
In one paragraph

Review in Frontiers in neurology, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing 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

1 citing paper in PubMed.

  1. 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

9 authors.

Karolina Ejsmont-SowałaMedical University of Bialystok, Bialystok, Poland.
Tomasz KsiążekMedical University of Bialystok, Bialystok, Poland.
Katarzyna Maciorowska-RosłanMedical University of Bialystok, Bialystok, Poland.
Joanna RosłanMedical University of Bialystok, Bialystok, Poland.
Agata CzarnowskaDepartment of Neurology, Medical University of Bialystok, Bialystok, Poland.
Anna Jakubiuk-TomaszukDepartment of Pediatric Neurology, Medical University of Bialystok, Bialystok, Poland.
Joanna TarasiukDepartment of Neurology, Medical University of Bialystok, Bialystok, Poland.
Katarzyna Kapica-TopczewskaDepartment of Neurology, Medical University of Bialystok, Bialystok, Poland.
Alina KułakowskaDepartment of Neurology, Medical University of Bialystok, Bialystok, Poland.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Xanthomatosis is a genetic disease inherited in an autosomal recessive manner. The specific phenotypic features are associated with patient's genetic profile. The result of the mutation is disorder of cholesterol synthesis and the accumulation of its precursors in tissues. The characteristic symptoms are progressive cerebellar ataxia, cataract, diarrhea, and the deposition of cholesterol in the tendons. Our objective is to follow-up information to treatment efficacy of 22-year-old patient diagnosed with cerebrotendinous xanthomatosis through 1.5 year observation. In 2012, an 11-year-old patient with a long history of deformed feet and frequent yellowing of the skin, was admitted to the Department of Neurology due to seizures. In 2013, the patient began to suffer from diarrhea, and its frequency was correlated with the concentration of bilirubin in the blood. In the same year cataract was diagnosed. Gradually, the patient starts to complain about progressive difficulties in moving. In 2019, genetic tests confirmed the diagnosis of cerebrotendinous xanthomatosis. Since July 2021, the patient has been treated with chenodeoxycholic acid. The deterioration of patient's mobility has been significantly inhibited, consequently his quality of life has improved. The presented case report underscores the efficacy of CDCA supplementation in halting the progression of CTX, resulting in marked improvements in the patient's quality of life.

Indexed as

CDCAcerebrotendinous xanthomatosischenodeoxycholic acidCTXCYP27A1

Identifiers

PMID38952472
PMCPMC11215067

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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.