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Intrathecal Baclofen Pump Therapy for Medically Refractory Stiff Person Syndrome: A Multicenter Case Series
Lior Ungar1, Sarah Hodges2, Suhrud Panchawagh3
1Department of Neurosurgery, Sheba Medical Center, Ramat Gan, Israel.
Summary
Intrathecal baclofen (ITB) therapy offers sustained relief for severe stiff person syndrome (SPS) patients unresponsive to other treatments. This approach improved stiffness, spasms, and function, demonstrating its potential for managing refractory SPS.
Area of Science:
- Neurology
- Pharmacology
Background:
- Stiff person syndrome (SPS) is a rare autoimmune disorder characterized by severe muscle rigidity and spasms.
- Medically refractory SPS often presents significant challenges in management, with limited treatment options.
- Intrathecal baclofen (ITB) therapy is an option for severe spasticity.
Purpose of the Study:
- To evaluate the clinical outcomes, optimal dosing, and safety profile of ITB therapy in patients with medically refractory SPS.
- To assess the long-term efficacy and durability of ITB treatment for severe SPS symptoms.
Main Methods:
- Retrospective review of six patients with anti-glutamic acid decarboxylase (GAD)-positive SPS.
- Analysis of clinical features, prior treatments, ITB dosing, complications, and functional outcomes post-implantation.
- Patients included those refractory to oral medications and immunotherapy.
Main Results:
- All patients experienced significant reductions in stiffness and spasm frequency.
- Improved mobility and respiratory function were observed in multiple patients.
- ITB therapy demonstrated sustained clinical benefit for 2.5 to four years, with one case of manageable septic shock.
Conclusions:
- Intrathecal baclofen (ITB) therapy is a viable and effective treatment for severe, medically refractory stiff person syndrome (SPS).
- Durable symptomatic improvement supports considering ITB in carefully selected SPS patients.
- ITB offers a promising therapeutic avenue for improving quality of life in refractory SPS.
