Evidence map›Paper›PMID 37564735›Full record

ReviewFrontiers in neurology2023

Adult-onset leukodystrophies: a practical guide, recent treatment updates, and future directions.

Karthik Muthusamy, Ajith Sivadasan, Luke Dixon, Sniya Sudhakar, Maya Thomas, Sumita Danda, Zbigniew K Wszolek, Klaas Wierenga, Radhika Dhamija, Ralitza Gavrilova

Open access · goldAbstract readReview
In one paragraph

Review in Frontiers in neurology, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 14 papers.

0numbers the graph read from it
0cells of the map it votes in
14citing papers in PubMed
4.2field-weighted citation impact, top 5% 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

14 citing papers in PubMed, 19 citations in OpenAlex.

  1. Review
  2. Article
  3. Article
  4. Article
  5. Characteristic MRI pattern inFrontiers in neuroscience · 2026
    Article
  6. Article
  7. Diagnosing Alexander disease in adults.Practical neurology · 2025
    Review
  8. Article
  9. Article
  10. Article
  11. Article
  12. Gene therapy for the leukodystrophies: From preclinical animal studies to clinical trials.Neurotherapeutics : the journal of the American Society for Experimental NeuroTherapeutics · 2024
    Review
  13. Review
  14. A novel mutation inBMJ case reports · 2023
    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

10 authors at 7 institutions in 3 countries.

Karthik MuthusamyDepartment of Clinical Genomics, Mayo Clinic, Jacksonville, FL, United States.
Ajith SivadasanDepartment of Neurological Sciences, Christian Medical College, Tamil Nadu, Vellore, India.
Luke DixonDepartment of Radiology, Imperial College, NHS Trust, London, United Kingdom.
Sniya SudhakarDepartment of Radiology, Great Ormond Street Hospital, London, United Kingdom.
Maya ThomasDepartment of Neurological Sciences, Christian Medical College, Tamil Nadu, Vellore, India.
Sumita DandaDepartment of Medical Genetics, Christian Medical College, Vellore, Tamil Nadu, India.
Zbigniew K WszolekDepartment of Neurology, Mayo Clinic, Jacksonville, FL, United States.
Klaas WierengaDepartment of Clinical Genomics, Mayo Clinic, Jacksonville, FL, United States.
Radhika DhamijaDepartment of Clinical Genomics and Neurology, Mayo Clinic, Phoenix, AZ, United States.
Ralitza GavrilovaDepartment of Clinical Genomics and Neurology, Mayo Clinic, Rochester, MN, United States.
Mayo Clinic in Florida · USChristian Medical College · INChristian Medical College & Hospital · INGreat Ormond Street Hospital · GBImperial College Healthcare NHS Trust · GBMayo Clinic · USWinnMed · US

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Adult-onset leukodystrophies though individually rare are not uncommon. This group includes several disorders with isolated adult presentations, as well as several childhood leukodystrophies with attenuated phenotypes that present at a later age. Misdiagnoses often occur due to the clinical and radiological overlap with common acquired disorders such as infectious, immune, inflammatory, vascular, metabolic, and toxic etiologies. Increased prevalence of non-specific white matter changes in adult population poses challenges during diagnostic considerations. Clinico-radiological spectrum and molecular landscape of adult-onset leukodystrophies have not been completely elucidated at this time. Diagnostic approach is less well-standardized when compared to the childhood counterpart. Absence of family history and reduced penetrance in certain disorders frequently create a dilemma. Comprehensive evaluation and molecular confirmation when available helps in prognostication, early initiation of treatment in certain disorders, enrollment in clinical trials, and provides valuable information for the family for reproductive counseling. In this review article, we aimed to formulate an approach to adult-onset leukodystrophies that will be useful in routine practice, discuss common adult-onset leukodystrophies with usual and unusual presentations, neuroimaging findings, recent advances in treatment, acquired mimics, and provide an algorithm for comprehensive clinical, radiological, and genetic evaluation that will facilitate early diagnosis and consider active treatment options when available. A high index of suspicion, awareness of the clinico-radiological presentations, and comprehensive genetic evaluation are paramount because treatment options are available for several disorders when diagnosed early in the disease course.

Indexed as

adult-onset leukodystrophyleukodystrophyleukoencephalopathyneurogeneticsneurometabolic disorder

Identifiers

PMID37564735
PMCPMC10410460
OpenAlexW4385272115

What OpenQuestion holds

Textmetadata
LicenceCC BY
Read underepoch 390

Registered trials

None linked

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.