The prevalence and clinical phenotype of dual seropositive neuromyelitis optica spectrum disorders at a national reference center in South Asia

Lalwani, Chirag Sunil; Faisal, Fida; Yadav, Anjali; Kannoth, Sudheeran; Nambiar, Vivek; Gopinath, Sibi; Kumar, Anand; Saraf, Udit; Vincent, Jino; Anoop, Sruthi; Mathai, A; Panicker, Suprabha

doi:10.1016/j.msard.2023.104736

Cited by 1 publication

(2 citation statements)

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“…Treatment with methylprednisolone was ineffective for all patients. However, plasmapheresis and/or periodic rituximab injections produced an excellent response 26 . Similar data for paraneoplastic NMOSD/MOGAD are not yet available, and further research is needed to investigate whether dual seropositivity influences outcome.…”

Section: Discussionmentioning

confidence: 95%

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Anti‐aquaporin‐4 immunoglobulin G/anti‐myelin oligodendrocyte glycoprotein immunoglobulin G double‐positive paraneoplastic neurological syndrome in a patient with triple‐negative breast cancer

Siddiqui,

Ross,

Jani

et al. 2023

Clinical & Exp Neuroim

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We report a rare case of paraneoplastic neurological syndrome with dual seropositivity of anti‐aquaporin‐4 and myelin oligodendrocyte glycoprotein antibodies in a 40 year‐old woman with metastatic triple‐negative breast cancer. She received multiple lines of anti‐neoplastic treatment, including immunotherapy with pembrolizumab, as well as cytotoxic chemotherapy. Paraneoplastic meningoencephalomyelitis developed 2 years after diagnosis of breast cancer and 1 year after discontinuation of immunotherapy with pembrolizumab. She first developed longitudinally extending transverse myelitis followed by left optic neuritis and meningoencephalitis with new enhancing lesions in the brain and spinal leptomeninges. Cerebrospinal fluid analysis during both episodes showed normal glucose and protein, and elevated white blood cell count. Cytology was negative for malignancy. Cerebrospinal fluid was positive for neuromyelitis optica immunoglobulin G antibody anti‐aquaporin‐4, and autoimmune myelopathy panel was positive for myelin oligodendrocyte glycoprotein antibody. The patient had significant clinical and radiographic improvement after completion of five cycles of plasmapheresis followed by intravenous immunoglobulin. She did not have recurrence of paraneoplastic syndrome with maintenance rituximab every 6 months and daily low‐dose prednisone. She succumbed to progressive systemic metastatic disease 4.5 years after her breast cancer diagnosis. This case shows that these antibodies can occur concurrently and cause clinical features, such as both neuromyelitis optica spectrum disorder and myelin oligodendrocyte glycoprotein antibody disease, in a patient with a singular type of cancer. We highlight the importance of testing for paraneoplastic etiology in cancer patients with radiographic menigoencephalomyelitis or meningitis with atypical symptoms of meningeal carcinomatosis and/or cerebrospinal fluid profile negative for leptomeningeal carcinomatosis.

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Section: Discussionmentioning

confidence: 95%

“…These double‐positive patients had a multiphase disease course with a high annual relapse rate and severe residual disability 24,25 . A study of 1935 cases of NMOSD showed dual‐positive (AQP4 antibody + MOG antibody) in three cases (0.15%) 26 . Its clinical phenotype was more like NMOSD rather than MOGAD.…”

Section: Discussionmentioning

confidence: 99%