ArticleMedicine2015
A Case of Stiff Person Syndrome: Immunomodulatory Effect of Benzodiazepines: Successful Rituximab and Tizanidine Therapy.
Article in Medicine, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 14 papers, 1 of them a synthesis that pooled it.
What it found
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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.
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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.
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Who cites it
14 citing papers in PubMed, 1 synthesis or guideline pooled it, 21 citations in OpenAlex.
- Efficacy of efgartigimod in the treatment of autoimmune encephalitis: a systematic review.Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology · 2026Pooled it
- Case report of stiff - person syndrome and literature review.IBRO neuroscience reports · 2026Article
- Long-term neurological consequences following benzodiazepine exposure: A scoping review.PloS one · 2025Article
- Stiff person syndrome in a Nepalese man with uncontrolled diabetes mellitus and ketonuria: A rare case report.Clinical case reports · 2023Article
- An update on malignant tumor-related stiff person syndrome spectrum disorders: clinical mechanism, treatment, and outcomes.Frontiers in neurology · 2023Review
- Therapeutic plasma exchange in the management of stiff person syndrome spectrum disorders: a case series and review of the literature.Therapeutic advances in neurological disorders · 2023Article
- Efficacy and safety of therapeutic plasma exchange in stiff person syndrome.Open medicine (Warsaw, Poland) · 2021Review
- Review
- Limited Utility for Benzodiazepines in Chronic Pain Management: A Narrative Review.Advances in therapy · 2020Review
- Stiff-person syndrome in a patient with comorbid bipolar and panic disorders: A case report and literature review.The mental health clinician · 2020Article
- Association of stiff-person syndrome with autoimmune endocrine diseases.World journal of clinical cases · 2019Article
- Evaluation of Treatment Outcomes in Patients with Stiff Person Syndrome with Rituximab vs. Standard of Care.Cureus · 2017Review
- Anti-B-Cell Therapies in Autoimmune Neurological Diseases: Rationale and Efficacy Trials.Neurotherapeutics : the journal of the American Society for Experimental NeuroTherapeutics · 2016Review
- Therapeutic role of plasma exchange in the management of stiff person syndrome: experience from a tertiary care centre.Hematology, transfusion and cell therapyArticle
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
1 author at 1 institution in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Stiff person syndrome (SPS) is a rare autoimmune disease. Most patients have high-titer antibodies against glutamate decarboxylase (GADAb), which is without practical value in disease monitoring. Benzodiazepines are the first line drugs, but long-term use is not well characterized. This report demonstrates ineffective benzodiazepine therapy of SPS that prompts tachyphylaxis, loss of responsiveness, and finally benzodiazepine withdrawal syndrome. Convulsion and anxiety correlate with high level of creatine phosphokinase (CK). Although tonus and spasm attacks were successfully controlled by tizanidine, glutamate release inhibitor, the immune response, and autoimmune diabetes development require the plasmapheresis, mycophenolat mofetil, and rituximab therapy that results in a significant decrease of GADAb, impaired glucose tolerance (IGT), lactate dehydrogenase (LDH), and CK normalization. Unfortunately, reintroduction of benzodiazepine was a source of rapid and high increase of CK, LDH, GADAb titer (up to 1:15,000), IGT, and SPS relapse. Contrary to previous publications, we observed IGT that correlated with high anti-GAD level, but without high immunogenetic susceptibility to haplotype human leukocyte antigens-DR3, DQw2. This preliminary observation and the last finding of immunomodulatory properties of peripheral benzodiazepine receptor suggest that increased antigenic stimulation during benzodiazepine therapy and glutamatergic hyperactivity could account for convulsions observed in SPS. Benzodiazepine withdrawal prompted alternative muscle relaxant therapy (tizanidine). Muscular and brain abnormalities observed in SPS indicate that noncardiac CK level may be a useful tool in SPS therapy monitoring.
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Registered trials
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.