脳障害と治療

概要

A Case of CANOMAD with Review of the Literature

Stephen Halpin, Samer Al-Hussayni, Sibte Hasan, Mark Busby and Rosaria Buccoliero*

Introduction: CANOMAD (an acronym for its full manifestation of chronic ataxic neuropathy, ophthalmoplegia, IgM paraprotein, cold agglutinins and disialosyl antibodies) is an uncommon paraproteinaemic neuropathy.

Methods and Results: We present the case of a man of 58 years at presentation, with chronic ataxic neuropathy, ophthalmoplegia, and an IgM paraprotein who was found to have antibodies active against disialosylgangliosides. Despite treatment trials with a range of immunosuppressive therapies, intravenous immunoglobulin proved the only effective treatment strategy in his case.

Conclusions: CANOMAD has been described in a growing number of individual reports and small series for which we have methodically searched the literature. In the absence of randomised trials of treatment for this disabling condition, we bring together these published clinical experiences, describing the degree of heterogeneity in the presentation/laboratory findings and demonstrating that intravenous immunoglobulin is the therapy found most frequently to be successful.