Evidence map›Paper›PMID 37658208›Full record

ArticleNeurogenetics2023

Adult-onset Alexander disease among patients of Jewish Syrian descent.

Saar Anis, Tsvia Fay-Karmon, Simon Lassman, Fadi Shbat, Orit Lesman-Segev, Nofar Mor, Ortal Barel, Dan Dominissini, Odelia Chorin, Elon Pras and 2 more

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Article in Neurogenetics, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
0.8field-weighted citation impact, top 27% 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

2 citing papers in PubMed, 5 citations in OpenAlex.

  1. Article
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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

12 authors at 3 institutions in 2 countries.

Saar AnisMovement Disorders Institute and Department of Neurology, Sheba Medical Center, Tel Hashomer, Ramat Gan, Israel.ORCID 0000-0002-6448-4505
Tsvia Fay-KarmonMovement Disorders Institute and Department of Neurology, Sheba Medical Center, Tel Hashomer, Ramat Gan, Israel.
Simon LassmanSt George's Hospital, University of London, London, UK.
Fadi ShbatMovement Disorders Institute and Department of Neurology, Sheba Medical Center, Tel Hashomer, Ramat Gan, Israel.
Orit Lesman-SegevFaculty of Medicine, Tel Aviv University, Tel Aviv, Israel.
Nofar MorCancer Research Center and Wohl Institute for Translational Medicine, Tel Hashomer, Ramat Gan, Israel.
Ortal BarelCancer Research Center and Wohl Institute for Translational Medicine, Tel Hashomer, Ramat Gan, Israel.
Dan DominissiniFaculty of Medicine, Tel Aviv University, Tel Aviv, Israel.
Odelia ChorinFaculty of Medicine, Tel Aviv University, Tel Aviv, Israel.
Elon PrasFaculty of Medicine, Tel Aviv University, Tel Aviv, Israel.
Lior GreenbaumFaculty of Medicine, Tel Aviv University, Tel Aviv, Israel.
Sharon Hassin-BaerMovement Disorders Institute and Department of Neurology, Sheba Medical Center, Tel Hashomer, Ramat Gan, Israel. Shassin@tauex.tau.ac.il.
Tel Aviv University · ILSheba Medical Center · ILUniversity of London · GB

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Alexander disease (AxD) is a rare autosomal dominant leukodystrophy caused by heterozygous mutations in the glial fibrillary acid protein (GFAP) gene. The age of symptoms onset ranges from infancy to adulthood, with variable clinical and radiological manifestations. Adult-onset AxD manifests as a chronic and progressive condition, characterized by bulbar, motor, cerebellar, and other clinical signs and symptoms. Neuroradiological findings typically involve the brainstem and cervical spinal cord. Adult-onset AxD has been described in diverse populations but is rare in Israel. We present a series of patients diagnosed with adult-onset AxD from three families, all of Jewish Syrian descent. Five patients (4 females) were diagnosed with adult-onset AxD due to the heterozygous mutation c.219G > A, p.Met73Ile in GFAP. Age at symptoms onset ranged from 48 to 61 years. Clinical characteristics were typical and involved progressive bulbar and gait disturbance, followed by pyramidal and cerebellar impairment, dysautonomia, and cognitive decline. Imaging findings included medullary and cervical spinal atrophy and mostly infratentorial white matter hyperintensities. A newly recognized cluster of adult-onset AxD in Jews of Syrian origin is presented. This disorder should be considered in differential diagnosis in appropriate circumstances. Genetic counselling for family members is required in order to discuss options for future family planning.

Indexed as

Alexander DiseaseAdultAtrophyFemaleGlial Fibrillary Acidic ProteinHumansJewsMiddle AgedMutationSyriaGlial Fibrillary Acidic ProteinAdult-onsetAlexander diseaseGFAPLeukodystrophy

Identifiers

PMID37658208
OpenAlexW4386360353

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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.