Relapsing sensorimotor neuropathy with ophthalmoplegia, antidisialosyl antibodies, and extramembranous glomerulonephritis

A 72‐year‐old man presented with oculomotor dysfunction, subacute relapsing sensorimotor neuropathy, elevated erythrocyte sedimentation rate, IgM monoclonal paraprotein, cold agglutinins, and antidisialosyl IgM antibodies, features previously described by the acronym CANOMAD (chronic ataxic neuropathy with ophthalmoplegia, M protein, agglutination, and disialosyl antibodies). The patient also had extramembranous glomerulopathy associated with this syndrome. Treatment with corticosteroids improved both the neuropathy and glomerulopathy. This case suggests that the spectrum of neuropathy associated with monoclonal gammopathy may be broader than originally believed. Muscle Nerve, 2006

[1]  T. Miyatake,et al.  Ataxic polyneuropathy and anti-Pr2 IgMκ M proteinemia , 1992, Journal of Neurology.

[2]  A. Steck,et al.  Motor neuropathy with activity of monoclonal IgM antibody to GD1a ganglioside , 1989, Journal of Neurology.

[3]  W. Schady,et al.  The clinical and laboratory features of chronic sensory ataxic neuropathy with anti-disialosyl IgM antibodies. , 2001, Brain : a journal of neurology.

[4]  R. Weinstein,et al.  Treatment experience in patients with anti–myelin‐associated glycoprotein neuropathy , 2001, Muscle & nerve.

[5]  R. Mobbs,et al.  Chronic inflammatory demyelinating polyneuropathy associated with membranous glomerulonephritis: case report , 2000, Journal of Clinical Neuroscience.

[6]  S. Kusunoki,et al.  Sensorimotor polyneuropathy associated with chronic lymphocytic leukemia, IgM antigangliosides antibody and human T‐cell leukemia virus I infection , 1999, Muscle & nerve.

[7]  M. Weiss,et al.  Autoantibodies associated with peripheral neuropathy , 1999, Muscle & nerve.

[8]  N. Baumann,et al.  Peripheral neuropathy associated with IgM monoclonal gammopathy: Correlations between M‐protein antibody activity and clinical/electrophysiological features in 40 cases , 1998, Muscle & nerve.

[9]  A. Miettinen,et al.  Decrease of glomerular disialogangliosides in puromycin nephrosis of the rat. , 1996, The American journal of pathology.

[10]  H. Willison,et al.  A somatically mutated human antiganglioside IgM antibody that induces experimental neuropathy in mice is encoded by the variable region heavy chain gene, V1-18. , 1996, The Journal of clinical investigation.

[11]  G. Eknoyan,et al.  Membranous glomerulonephritis associated with inflammatory demyelinating peripheral neuropathies. , 1996, American journal of kidney diseases : the official journal of the National Kidney Foundation.

[12]  L. Illis,et al.  Monoclonal IgM Cold Agglutinins with Anti‐Pr1d Specificity in a Patient with Peripheral Neuropathy , 1994, Vox sanguinis.

[13]  A. Miettinen,et al.  Nephron segment and cell-type specific expression of gangliosides in the developing and adult kidney. , 1994, Kidney international.

[14]  S. Kusunoki,et al.  IgM M‐protein in a patient with sensory‐dominant neuropathy binds preferentially to polysialogangliosides , 1992, Acta neurologica Scandinavica.

[15]  T. Miyatake,et al.  Ataxic polyneuropathy and anti-Pr2 IgM kappa M proteinemia. , 1992, Journal of neurology.

[16]  A. Pestronk,et al.  Invited review: motor neuropathies, motor neuron disorders, and antiglycolipid antibodies. , 1991, Muscle & nerve.

[17]  J. Burke,et al.  Membranous glomerulonephritis associated with chronic progressive demyelinating neuropathy , 1987, Neurology.

[18]  R. Brady,et al.  Monoclonal IgM in a patient with paraproteinemic polyneuropathy binds to gangliosides containing disialosyl groups , 1985, Annals of neurology.

[19]  N. Baumann,et al.  Direct sensitive immunocharacterization of gangliosides on plastic thin-layer plates using peroxidase staining. , 1985, Journal of immunological methods.

[20]  T. Feizi,et al.  The reactivities of human erythrocyte autoantibodies anti-Pr2, anti-Gd, Fl and Sa with gangliosides in a chromatogram binding assay. , 1984, The Biochemical journal.

[21]  N. Latov,et al.  Myelin‐Associated Glycoprotein Is the Antigen for a Monoclonal IgM in Polyneuropathy , 1982, Journal of neurochemistry.

[22]  R. McCluer,et al.  The Use of Sep‐Pak™ C18 Cartridges During the Isolation of Gangliosides , 1980, Journal of neurochemistry.

[23]  H. Seelig,et al.  Identification of I/i, Pr1-3 and Gd antigens in the human kidney: possible relevance to hyperacute graft rejection induced by cold agglutinins. , 1978, Clinical and experimental immunology.

[24]  J. Folch,et al.  A simple method for the isolation and purification of total lipides from animal tissues. , 1957, The Journal of biological chemistry.