“…Anti-GD2 monoclonal antibody reacted with the myelin sheaths in the peripheral nerves as well as with the pituicyte cytoplasm in the posterior lobe of the pituitary gland [10,11]. Iorio et al [12] reported a patient who developed SIADH during the first relapse chronic ataxic neuropathy with ophthalmoplegia, IgM paraprotein, cold agglutinins, and disialosyl antibodies. The dysregulation of osmolarity receptors may be consequent to binding of some anti-disialosyl antibodies to specific hypothalamic epitopes [12].…”
Section: Discussionmentioning
confidence: 99%
“…Iorio et al [12] reported a patient who developed SIADH during the first relapse chronic ataxic neuropathy with ophthalmoplegia, IgM paraprotein, cold agglutinins, and disialosyl antibodies. The dysregulation of osmolarity receptors may be consequent to binding of some anti-disialosyl antibodies to specific hypothalamic epitopes [12]. The elevation of anti-GD1b, -GQ1b, and -GT1a antibodies may be pathologically related to SIADH.…”
We report a 72-year-old woman with Miller-Fisher syndrome (MFS) with syndrome of inappropriate secretion of antidiuretic hormone (SIADH). She developed diplopia and unsteady gait a week after an upper respiratory infection. Neurologic examination revealed ophthalmoplegia, ataxia, symmetrical weakness, numbness, and areflexia. She underwent intravenous immunoglobulin therapy. Her serum sodium concentration decreased to 119 mEq/L on day 12. She had low plasma osmolarity (254 mosm/kg), high urine osmolarity (457 mosm/kg), and high urine sodium level (73 mEq/L), while the blood level of antidiuretic hormone was normal. Anti-GD1b immunoglobulin G (IgG), -GQ1b IgG, -GT1a IgG, and -Gal-C IgM antibodies were positive. We diagnosed her with MFS overlapping with SIADH. Four weeks after onset, her symptoms recovered. The elevation of anti-GD1b, -GQ1b, and -GT1a antibodies that recognize disialosyl residue may be pathologically related to SIADH.
“…Anti-GD2 monoclonal antibody reacted with the myelin sheaths in the peripheral nerves as well as with the pituicyte cytoplasm in the posterior lobe of the pituitary gland [10,11]. Iorio et al [12] reported a patient who developed SIADH during the first relapse chronic ataxic neuropathy with ophthalmoplegia, IgM paraprotein, cold agglutinins, and disialosyl antibodies. The dysregulation of osmolarity receptors may be consequent to binding of some anti-disialosyl antibodies to specific hypothalamic epitopes [12].…”
Section: Discussionmentioning
confidence: 99%
“…Iorio et al [12] reported a patient who developed SIADH during the first relapse chronic ataxic neuropathy with ophthalmoplegia, IgM paraprotein, cold agglutinins, and disialosyl antibodies. The dysregulation of osmolarity receptors may be consequent to binding of some anti-disialosyl antibodies to specific hypothalamic epitopes [12]. The elevation of anti-GD1b, -GQ1b, and -GT1a antibodies may be pathologically related to SIADH.…”
We report a 72-year-old woman with Miller-Fisher syndrome (MFS) with syndrome of inappropriate secretion of antidiuretic hormone (SIADH). She developed diplopia and unsteady gait a week after an upper respiratory infection. Neurologic examination revealed ophthalmoplegia, ataxia, symmetrical weakness, numbness, and areflexia. She underwent intravenous immunoglobulin therapy. Her serum sodium concentration decreased to 119 mEq/L on day 12. She had low plasma osmolarity (254 mosm/kg), high urine osmolarity (457 mosm/kg), and high urine sodium level (73 mEq/L), while the blood level of antidiuretic hormone was normal. Anti-GD1b immunoglobulin G (IgG), -GQ1b IgG, -GT1a IgG, and -Gal-C IgM antibodies were positive. We diagnosed her with MFS overlapping with SIADH. Four weeks after onset, her symptoms recovered. The elevation of anti-GD1b, -GQ1b, and -GT1a antibodies that recognize disialosyl residue may be pathologically related to SIADH.
“…Earlier described CANOMAD cases reported patients with optic nerve involvement, extramembranous glomerulonephritis, and a syndrome of inappropriate antidiuretic hormone secretion (SIADH) 2,[11][12][13][14] . Cases without ophthalmoplegia, fur thermore, with temporary respiratory failure associated with facial involuntary movements were also mentioned 2,[11][12][13][14] . We report a case of CANO -MAD syndrome with severe respiratory failure followed by neuropathological evaluation.…”
“…The diagnosis of Waldenstrom macroglobulinemia only requires demonstration of a serum IgM monoclonal pararotein with histological evidence of bone marrow infiltration by lymphoplasmacytic cells, without a minimal serum IgM level or minimal percentage of bone marrow infiltration . However, bone marrow examination was not reported or performed in patients from the largest clinical series of 18 cases or in case reports of a further 6 patients Three patients reported with CANOMAD had a negative bone marrow, and 1 autopsy showed 10–15% plasma cells in the bone marrow with no comment regarding light chain restriction or IgH clonality studies . Our patient underwent bone marrow examination due to the elevated serum IgM (15 g/L), mild anemia, and associated neuropathy.…”
The autopsy provides evidence of the pathogenesis of the relapsing remitting component of CANOMAD, and we postulate that this may relate to the presence of clonal IgM anti-disiayl gangliosides secreting B-lymphocytes within nerves.
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