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Anesth Analg 2007;104:438-441
© 2007 International Anesthesia Research Society
doi: 10.1213/01.ane.0000252965.83347.25

ANALGESIA

Defining a Treatable Cause of Erythromelalgia: Acute Adolescent Autoimmune Small-Fiber Axonopathy

Joshua Paticoff, MD*, Assia Valovska, MD*, Srdjan S. Nedeljkovic, MD*, and Anne Louise Oaklander, MD, PhD{dagger}

From the *Department of Anesthesiology, Perioperative and Pain Medicine, Brigham and Women's Hospital; and {dagger}Departments of Neurology and Pathology (Neuropathology), Massachusetts General Hospital, Harvard Medical School, Boston, Massachusetts.

Abstract

Conditions described as "erythromelalgia" and "erythermalgia" are being formally specified by etiological diagnoses that enable the use of disease-modifying as well as symptomatic treatments. We describe an otherwise healthy 20-year-old man with acute-onset erythromelalgia. Severe bilateral distal limb pain and vasodilation persisted despite the use of many antihyperalgesics. Pathological examination of cutaneous nerve endings revealed severe small-fiber predominant axonopathy. Treatment of his apparent autoimmune polyneuropathy with high dose corticosteroids, 4 days of lidocaine infusion, and a prednisone taper cured him. Similarities to other cases allowed us to tentatively characterize a new treatable cause of erythromelalgia; acute adolescent autoimmune small-fiber axonopathy. In this report we evaluate various options for diagnosis and treatment.






Lippincott, Williams & Wilkins Anesthesia & Analgesia® is published for the International Anesthesia Research Society® by Lippincott Williams & Wilkins with the assistance of Stanford University Libraries' HighWire Press®. Copyright 2006 by the International Anesthesia Research Society. Online ISSN: 1526-7598   Print ISSN: 0003-2999 HighWire Press
Copyright © 2007 by the International Anesthesia Research Society.