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Frequency of myelin oligodendrocyte glycoprotein antibodies in pediatric onset multiple sclerosis

  • Author Footnotes
    1 Present address: Faculty of Medicine, Department of Pediatric Neurology, Dr. Sami Ulus Maternity and Child Health and Diseases Training and Research Hospital, University of Health Sciences, Ankara 06080, Turkey.
    Ismail Solmaz
    Correspondence
    Corresponding author.
    Footnotes
    1 Present address: Faculty of Medicine, Department of Pediatric Neurology, Dr. Sami Ulus Maternity and Child Health and Diseases Training and Research Hospital, University of Health Sciences, Ankara 06080, Turkey.
    Affiliations
    Faculty of Medicine, Department of Pediatric Neurology, Hacettepe University, Ankara, Turkey
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  • Tansu Doran
    Affiliations
    Koç University Research Center for Translational Medicine, Istanbul, Turkey
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  • Mohammadreza Yousefi
    Affiliations
    Koç University Research Center for Translational Medicine, Istanbul, Turkey
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  • Author Footnotes
    1 Present address: Faculty of Medicine, Department of Pediatric Neurology, Dr. Sami Ulus Maternity and Child Health and Diseases Training and Research Hospital, University of Health Sciences, Ankara 06080, Turkey.
    Bahadır Konuskan
    Footnotes
    1 Present address: Faculty of Medicine, Department of Pediatric Neurology, Dr. Sami Ulus Maternity and Child Health and Diseases Training and Research Hospital, University of Health Sciences, Ankara 06080, Turkey.
    Affiliations
    Faculty of Medicine, Department of Pediatric Neurology, Hacettepe University, Ankara, Turkey
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  • Ibrahim Oncel
    Affiliations
    Faculty of Medicine, Department of Pediatric Neurology, Hacettepe University, Ankara, Turkey
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  • Atay Vural
    Affiliations
    Koç University Research Center for Translational Medicine, Istanbul, Turkey

    Department of Neurology, Koç University, Istanbul, Turkey
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  • Banu Anlar
    Affiliations
    Faculty of Medicine, Department of Pediatric Neurology, Hacettepe University, Ankara, Turkey
    Search for articles by this author
  • Author Footnotes
    1 Present address: Faculty of Medicine, Department of Pediatric Neurology, Dr. Sami Ulus Maternity and Child Health and Diseases Training and Research Hospital, University of Health Sciences, Ankara 06080, Turkey.
Published:August 08, 2022DOI:https://doi.org/10.1016/j.msard.2022.104097

      Highlights

      • MOG-Ab associated disease is more common in children than in adults.
      • We examined MOG-Ab status in our pediatric multiple sclerosis (pMS) cases.
      • MOG-Ab positivity in our pMS series was similar to rates reported in adult MS.
      • Relapse rate was low and course was similar in MOG-Ab positive and negative pMS cases.

      Abstract

      Background

      Myelin oligodendrocyte glycoprotein antibodies (MOG-Ab) are associated with acute demyelinating syndromes and only rarely detected in multiple sclerosis (MS). As MOG-Ab associated disease is common in childhood, we speculated young patients might be more likely to produce MOG-Ab and investigated the frequency of MOG-Ab seropositivity in pediatric onset MS (POMS).

      Material and methods

      Patients who experienced their first acute demyelinating event before age 18 years and were diagnosed with MS during follow-up were included in this single-center study. Patient data were retrieved from clinical records. Serum samples obtained and frozen at clinical visits were analyzed for MOG-Ab by a live cell-based assay (CBA) measuring delta mean fluorescence intensity (MFI) and MFI ratio. The control group consisted of patients referred to pediatric neurology for headache or vertigo and who had no neurological disorder (n = 48). Another control group consisted of patients with systemic inflammatory disorders systemic lupus erythematosus (n = 17) and juvenile idiopathic arthritis (n = 13) diagnosed in the rheumatology clinic.

      Results

      The patient group (n = 122, F/M: 90/32, mean age 17.8 ± 2.6 years) were initially diagnosed as: MS, 62/122 (50.8%), clinically isolated syndrome, 43/122 (35.2%), radiologically isolated syndrome, 9/122 (7.3%), and acute disseminated encephalomyelitis 8/122 (6.5%). All received the final diagnosis of POMS. Serum was sampled 22.4 ± 29.2 (0–132) months after the first episode. None of the control groups had MOG-Ab positivity while 2/122 (1.6%) POMS cases had MOG-Abs, and a third patient had positive MFI and a MFI ratio slightly below the cut-off. These three patients’ initial and final diagnoses were MS, their annualized relapsing rates (ARRs) were 0.4–0.6, and most recent Expanded Disability Status Scale was 0.

      Conclusion

      Low titers of MOG-Ab can be detected in a small number of POMS patients at similar frequency with adult MS. Our POMS cases with MOG-Abs presented brainstem-cerebellar findings or seizures and had low ARR. Further series and longer follow-up will define whether these cases differ significantly from MOG-Ab negative POMS cases.

      Keywords

      Abbreviations:

      MS (multiple sclerosis), HC (healthy control), JIA (juvenile idiopathic arthritis), SLE (systemic lupus erythematosus), MFI (mean of fluorescence intensity), MOG-Ab (myelin oligodendrocyteglycoprotein antibody)
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