Literature DB >> 24722712

CANOMAD responding to weekly treatment with intravenous immunoglobulin (IVIg).

Martin Krenn1, Geoff Keir, Udo Carl Wieshmann.   

Abstract

A 48-year-old man presented with numbness in fingers and diplopia 1 week after a flu-like illness. He made a full recovery but 8 years later developed progressive and disabling sensory ataxia. He had superimposed acute flare-ups with numbness, double vision and ptosis, all following infections. A blood test showed antidisialosyl antibodies including GD1b, GD3, GT1b and GQ1b in keeping with the diagnosis of chronic ataxic neuropathy, ophthalmoplegia, IgM paraprotein, cold agglutinins and antidisialosyl antibodies (CANOMAD). Initial treatment with monthly courses of intravenous immunoglobulin (IVIg) 0.4 g/kg/day for 5 days every 4 weeks helped temporarily but there were marked disabling fluctuations of symptoms. With IVIg 0.6 g/kg/day weekly his symptoms are stable. He remains mobile and has no eye symptoms without need for any other medication. This case demonstrates that weekly IVIg infusions instead of one 5-day course monthly may be able to avoid fluctuations of symptoms in CANOMAD.

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Year:  2014        PMID: 24722712      PMCID: PMC3987528          DOI: 10.1136/bcr-2013-202545

Source DB:  PubMed          Journal:  BMJ Case Rep        ISSN: 1757-790X


  10 in total

1.  Chronic ataxic neuropathy with cold agglutinins: atypical phenotype and response to anti-CD20 antibodies.

Authors:  K Siddiqui; E Cahalane; M Keogan; O Hardiman
Journal:  Neurology       Date:  2003-11-11       Impact factor: 9.910

2.  Canomad presenting without ophthalmoplegia and responding to intravenous immunoglobulin.

Authors:  Cheryl Kam; Michelle S Balaratnam; Alistair Purves; Kerry R Mills; Paul Riordan-Eva; Steve Pollock; Istvan Bodi; Neil A R Munro; David L H Bennett
Journal:  Muscle Nerve       Date:  2011-11       Impact factor: 3.217

3.  Successful treatment with rituximab of one patient with CANOMAD neuropathy.

Authors:  Emilien Delmont; P Y Jeandel; A M Hubert; L Marcq; J Boucraut; C Desnuelle
Journal:  J Neurol       Date:  2009-12-04       Impact factor: 4.849

Review 4.  Intravenous immune globulin in autoimmune and inflammatory diseases.

Authors:  Erwin W Gelfand
Journal:  N Engl J Med       Date:  2012-11-22       Impact factor: 91.245

5.  Successful treatment of CANOMAD with IVIg and rituximab.

Authors:  Wolfgang N Löscher; Alexander Woertz; Monika Wallnöfer; Julia V Wanschitz; Gerhard Luef
Journal:  J Neurol       Date:  2013-02-12       Impact factor: 4.849

6.  The clinical and laboratory features of chronic sensory ataxic neuropathy with anti-disialosyl IgM antibodies.

Authors:  H J Willison; C P O'Leary; J Veitch; L D Blumhardt; M Busby; M Donaghy; P Fuhr; H Ford; A Hahn; S Renaud; H A Katifi; S Ponsford; M Reuber; A Steck; I Sutton; W Schady; P K Thomas; A J Thompson; J M Vallat; J Winer
Journal:  Brain       Date:  2001-10       Impact factor: 13.501

7.  Clinimetric evaluation of a new overall disability scale in immune mediated polyneuropathies.

Authors:  I S J Merkies; P I M Schmitz; F G A van der Meché; J P A Samijn; P A van Doorn
Journal:  J Neurol Neurosurg Psychiatry       Date:  2002-05       Impact factor: 10.154

Review 8.  Rituximab in patients with chronic inflammatory demyelinating polyradiculoneuropathy: a report of 13 cases and review of the literature.

Authors:  L Benedetti; C Briani; D Franciotta; R Fazio; I Paolasso; C Comi; M Luigetti; M Sabatelli; F Giannini; G L Mancardi; A Schenone; E Nobile-Orazio; D Cocito
Journal:  J Neurol Neurosurg Psychiatry       Date:  2010-07-16       Impact factor: 10.154

9.  Monoclonal IgM in a patient with paraproteinemic polyneuropathy binds to gangliosides containing disialosyl groups.

Authors:  A A Ilyas; R H Quarles; M C Dalakas; P H Fishman; R O Brady
Journal:  Ann Neurol       Date:  1985-12       Impact factor: 10.422

10.  Variability in intravenous immunoglobulin G regimens for autoimmune neuromuscular disorders.

Authors:  Randy Broyles; Linda Rodden; Patty Riley; Melvin Berger
Journal:  Postgrad Med       Date:  2013-03       Impact factor: 3.840

  10 in total
  2 in total

Review 1.  Sustained response to subcutaneous immunoglobulins in chronic ataxic neuropathy with anti-disialosyl IgM antibodies (CANDA): report of two cases and review of the literature.

Authors:  D Marastoni; L Africa; G M Fabrizi; F Giannini; A Peretti; S Bocci; L Insana; S Ferrari; F Ginanneschi; G Zanette
Journal:  J Neurol       Date:  2020-04-28       Impact factor: 4.849

2.  Facial Involuntary Movements and Respiratory Failure in CANOMAD, Responsive to IVIG Therapy.

Authors:  Kate Johnson; Ashish Malkan; Mohamed Shaffi
Journal:  Case Rep Med       Date:  2015-11-30
  2 in total

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