TY - JOUR
T1 - Long-lasting pain and somatosensory disturbances in children with myelin oligodendrocyte glycoprotein antibody-associated disease
AU - Ichimiya, Yuko
AU - Chong, Pin Fee
AU - Sonoda, Yuri
AU - Tocan, Vlad
AU - Watanabe, Mitsuru
AU - Torisu, Hiroyuki
AU - Kira, Ryutaro
AU - Takahashi, Toshiyuki
AU - Kira, Jun Ichi
AU - Isobe, Noriko
AU - Sakai, Yasunari
AU - Ohga, Shouichi
N1 - Funding Information:
We thank all the patients and parents for cooperatively participating in the study. We also thank Dr. Toshiro Hara (President, Fukuoka Children’s Hospital) and laboratory members for their helpful discussions.
Funding Information:
This study was supported by JSPS KAKENHI grant numbers JP17K16271 (Ichimiya), JP22K07893 (Sonoda), JP19K10613 (Chong), and JP21K07464 (Isobe); research grants from the Ministry of Health, Labour and Welfare of Japan (JP22HA1003: Chong, JP21FC1005 and JP20FC1054: Sakai); a research grant on Intractable Diseases (Neuroimmunological Diseases) from the Ministry of Health, Labour and Welfare of Japan (JP20FC1030: Isobe); AMED under the grant number JP20ek0109411, JP20wm0325002h (Sakai) and JP21zf0127004 (Isobe); The Japan Epilepsy Research Foundation, and Kawano Masanori Memorial Public Interest Incorporated Foundation for Promotion of Pediatrics (Sakai).
Publisher Copyright:
© 2023, The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature.
PY - 2023/7
Y1 - 2023/7
N2 - Myelin oligodendrocyte glycoprotein antibody (MOG-Ab) is an autoantibody associated with acquired demyelinating syndrome (ADS) in childhood and adults. The pathogenic roles of MOG-Ab and long-term outcomes of children with MOG-Ab-associated disease (MOGAD) remain elusive. We investigated the clinical features of children with ADS during follow-up in our institute. Clinical data were retrospectively analyzed using medical charts of patients managed in Kyushu University Hospital from January 1st, 2001, to March 31st, 2022. Participants were children of < 18 years of age when they received a diagnosis of ADS in our hospital. Cell-based assays were used to detect MOG-Ab in serum or cerebrospinal fluid at the onset or recurrence of ADS. The clinical and neuroimaging data of MOG-Ab-positive and MOG-Ab-negative patients were statistically analyzed. Among 31 patients enrolled in this study, 22 (13 females, 59%) received tests for MOG antibodies. Thirteen cases (59%) were MOG-Ab-positive and were therefore defined as MOGAD; 9 (41%) were MOG-Ab-negative. There were no differences between MOGAD and MOG-Ab-negative patients in age at onset, sex, diagnostic subcategories, or duration of follow-up. MOGAD patients experienced headache and/or somatosensory symptoms more frequently than MOG-Ab-negative patients (12/13 (92%) vs. 3/9 (22%); p = 0.0066). Somatosensory problems included persistent pain with hyperesthesia in the left toe, perineal dysesthesia, and facial hypesthesia. No specific neuroimaging findings were associated with MOGAD or the presence of somatosensory symptoms. Conclusions: Long-lasting somatosensory disturbances are prominent comorbidities in children with MOGAD. Prospective cohorts are required to identify molecular and immunogenetic profiles associated with somatosensory problems in MOGAD. What is Known: • Recurrence of demyelinating events occurs in a group of children with myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD). What is New: • Long-lasting headache and somatosensory problems are frequent comorbidities with pediatric MOGAD. Pain and somatosensory problems may persist for more than 5 years. • Neuroimaging data do not indicate specific findings in children with somatic disturbances.
AB - Myelin oligodendrocyte glycoprotein antibody (MOG-Ab) is an autoantibody associated with acquired demyelinating syndrome (ADS) in childhood and adults. The pathogenic roles of MOG-Ab and long-term outcomes of children with MOG-Ab-associated disease (MOGAD) remain elusive. We investigated the clinical features of children with ADS during follow-up in our institute. Clinical data were retrospectively analyzed using medical charts of patients managed in Kyushu University Hospital from January 1st, 2001, to March 31st, 2022. Participants were children of < 18 years of age when they received a diagnosis of ADS in our hospital. Cell-based assays were used to detect MOG-Ab in serum or cerebrospinal fluid at the onset or recurrence of ADS. The clinical and neuroimaging data of MOG-Ab-positive and MOG-Ab-negative patients were statistically analyzed. Among 31 patients enrolled in this study, 22 (13 females, 59%) received tests for MOG antibodies. Thirteen cases (59%) were MOG-Ab-positive and were therefore defined as MOGAD; 9 (41%) were MOG-Ab-negative. There were no differences between MOGAD and MOG-Ab-negative patients in age at onset, sex, diagnostic subcategories, or duration of follow-up. MOGAD patients experienced headache and/or somatosensory symptoms more frequently than MOG-Ab-negative patients (12/13 (92%) vs. 3/9 (22%); p = 0.0066). Somatosensory problems included persistent pain with hyperesthesia in the left toe, perineal dysesthesia, and facial hypesthesia. No specific neuroimaging findings were associated with MOGAD or the presence of somatosensory symptoms. Conclusions: Long-lasting somatosensory disturbances are prominent comorbidities in children with MOGAD. Prospective cohorts are required to identify molecular and immunogenetic profiles associated with somatosensory problems in MOGAD. What is Known: • Recurrence of demyelinating events occurs in a group of children with myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD). What is New: • Long-lasting headache and somatosensory problems are frequent comorbidities with pediatric MOGAD. Pain and somatosensory problems may persist for more than 5 years. • Neuroimaging data do not indicate specific findings in children with somatic disturbances.
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U2 - 10.1007/s00431-023-04989-z
DO - 10.1007/s00431-023-04989-z
M3 - Article
C2 - 37119299
AN - SCOPUS:85153938588
SN - 0340-6199
VL - 182
SP - 3175
EP - 3185
JO - European Journal of Pediatrics
JF - European Journal of Pediatrics
IS - 7
ER -