Evidence map›Paper›PMID 42171443›Full record

ArticleEuropean journal of neurology2026

Long-Term Outcomes in Stiff Person Spectrum Disorder.

Georgios Mangioris, Andrew McKeon, James H Bower, Nisa Vorasoot, Divyanshu Dubey, Eoin P Flanagan, Sean J Pittock, Anastasia Zekeridou

Abstract read
In one paragraph

Article in European journal of neurology, 2026. 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
–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

2 citing papers in PubMed.

  1. Article
  2. Review
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.

Georgios MangiorisDepartment of Laboratory Medicine and Pathology, Mayo Clinic College of Medicine, Rochester, Minnesota, USA.
Andrew McKeonDepartment of Laboratory Medicine and Pathology, Mayo Clinic College of Medicine, Rochester, Minnesota, USA.
James H BowerDepartment of Neurology, Mayo Clinic College of Medicine, Rochester, Minnesota, USA.
Nisa VorasootDepartment of Laboratory Medicine and Pathology, Mayo Clinic College of Medicine, Rochester, Minnesota, USA.ORCID https://orcid.org/0000-0002-0751-8967
Divyanshu DubeyDepartment of Laboratory Medicine and Pathology, Mayo Clinic College of Medicine, Rochester, Minnesota, USA.
Eoin P FlanaganDepartment of Laboratory Medicine and Pathology, Mayo Clinic College of Medicine, Rochester, Minnesota, USA.
Sean J PittockDepartment of Laboratory Medicine and Pathology, Mayo Clinic College of Medicine, Rochester, Minnesota, USA.
Anastasia ZekeridouDepartment of Laboratory Medicine and Pathology, Mayo Clinic College of Medicine, Rochester, Minnesota, USA.ORCID https://orcid.org/0000-0002-1960-9065

Funding

Seronegative Autoimmune Encephalopathies: Biomarker Discovery, Validation & Deep PhenotypingR01NS126227 · NINDS · MAYO CLINIC ROCHESTER · PI Andrew McKeon · 2022 to 2026
$1.9M
NINDS NIH HHS R01 NS126227
6 · The paper itself

Abstract

backgroundLong-term outcome data for stiff person spectrum disorder (SPSD) remain limited and sustained response to treatment varies.

methodsIn this retrospective cohort study, we included patients with probable or definite SPSD (Mayo Clinic criteria), evaluated at Mayo Clinic (12/1995-07/2024) with ≥ 5 years of follow-up after diagnosis. Primary outcome measures included the modified Rankin scale (mRS) and use of gait aids.

resultsFifty-four patients were included, 41 (76%) were female; median age at onset was 45 years (16-78). Most were antibody-positive (51 [94%]), with high-titer (serum, ≥ 20 nmol/L) GAD65-IgG in 45 (83%). Nine patients had a stiff-limb presentation (17%). The median interval from onset to diagnosis was 2 years (0-34), and median follow-up was 9 years (5-26). Most patients (46 [85%]) received immune therapy; 37 (82%) reported improvement. All patients had significant impairment (mRS ≥ 2) at nadir, with 31 (57%) losing functional independence (mRS ≥ 3) and 40 (75%) requiring bilateral gait assistance. The median time from onset to nadir was 6 years (0-39). At last follow-up, most (37 [69%]) were functionally independent (mRS ≤ 2), including 8 (15%) with minimal restrictions (mRS ≤ 1). Bilateral gait assistance was needed in 22 (41%). Among 40 patients with ≥ 10 years of follow-up, 22 (56%) had an mRS ≤ 2 and 25 (63%) required a gait aid at last follow-up. Poor outcome in this population was associated with bilateral gait assistance at nadir and older age (p < 0.05).

conclusionsAlthough over half of SPSD patients lose functional independence at disease nadir, many regain independence and achieve well-controlled symptoms.

Indexed as

Stiff-Person SyndromeAdolescentAdultAgedAutoantibodiesFemaleFollow-Up StudiesGlutamate DecarboxylaseHumansMaleMiddle AgedRetrospective StudiesTreatment OutcomeYoung AdultAutoantibodiesGlutamate Decarboxylaseamphiphysin antibodyGAD‐65glutamic acid decarboxylase 65‐kilodalton isoform antibodyglycine receptor antibodystiff‐man syndrome

Identifiers

PMID42171443
PMCPMC13238517

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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.