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Relapsing-Remitting Immunotherapy Responsive Small-Fiber Neuropathy: Longitudinal Tracking Through 10 Years Including

Anne Louise Oaklander¹, Julia Allen¹, Nadja Dietliker¹

¹From the Department of Neurology (A.L.O.), Massachusetts General Hospital and Harvard Medical School; Department of Pathology (Neuropathology) (A.L.O.), Massachusetts General Hospital; Harvard T.H. Chan School of Public Health & Harvard Medical School (J.A.), Boston; Department of Medical Oncology and Hematology (N.D.), University Hospital Zurich; Department of Neurology (E.P.W.-S.), University of Bern; and Department Neruology (Neurozentrum) (E.P.W.-S.), Luzerner Kantonsspital, Switzerland.

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Summary

This summary is machine-generated.

This study tracked a woman with small fiber polyneuropathy (SFN) over five years, revealing a relapsing-remitting course. Multiple immunotherapies were effective, even during pregnancy and breastfeeding.

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Area of Science:

Neurology
Immunology

Background:

Small fiber polyneuropathy (SFN) is a condition affecting small nerve fibers, often with unclear causes.
Understanding the long-term course and treatment responses in SFN is crucial for patient management.

Purpose of the Study:

To investigate the pathogenesis, clinical presentation, and treatment efficacy in a case of initially idiopathic SFN.
To characterize the disease course and response to various immunotherapies over an extended period.

Main Methods:

A single patient with biopsy-confirmed idiopathic SFN was longitudinally monitored using validated metrics.
Disease course and responses to treatments including prednisone, immunoglobulins, rituximab, daratumumab, belimumab, and plasmapheresis were tracked over five years.

Main Results:

Viral infections triggered recurrent episodes of erythromelalgia (burning pain) in the hands, feet, and face.
The patient experienced a relapsing-remitting course, with flares responding to different immunotherapies, including during pregnancy and postpartum.
Successful management was achieved with multiple treatments, allowing for a healthy pregnancy and sustained remission during breastfeeding.

Conclusions:

The observed relapsing-remitting pattern and response to multiple immunotherapies suggest a dysimmune etiology for this SFN case.
Validated metrics were instrumental in defining the disease course and evaluating treatment effectiveness, particularly in complex scenarios like pregnancy.