Myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) is an inflammatory disorder of the central nervous system. Here, we are the first to report a MOGAD patient with hypersomnolence caused by bilateral hypothalamic lesions.
Case presentation
The case involved a 51-year-old female patient with MOGAD who showed hypersomnolence. She had been diagnosed with MOGAD at 50 years old and admitted due to the development of optic neuritis and encephalitis while under corticosteroid and immunosuppressant treatment. These were improved after the introduction of intravenous methylprednisolone and plasma exchange. The orexin level in the cerebrospinal fluid (CSF) was decreased to 142.2 pg/mL (<200 pg/mL) upon admission and improved to a normal level (298.3 pg/mL) after immunotherapy. Bilateral hypothalamic lesions and reduction of the orexin level in the CSF were considered associated with hypersomnia.
Conclusion
The orexin level in CSF was useful to monitor hypothalamic dysfunction in a patient with MOGAD.
{"title":"MOG-antibody-associated disorder with hypothalamic lesions associated with hypersomnia and decrease of orexin in CSF: A case report","authors":"Kanako Menjo, Shinji Ashida, Shohei Murata, Eijirou Tanaka, Chihiro Fujii, Masami Tanaka, Keiko Tanaka, Takashi Kanbayashi, Toshiki Mizuno","doi":"10.1111/cen3.12728","DOIUrl":"10.1111/cen3.12728","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Background</h3>\u0000 \u0000 <p>Myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) is an inflammatory disorder of the central nervous system. Here, we are the first to report a MOGAD patient with hypersomnolence caused by bilateral hypothalamic lesions.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Case presentation</h3>\u0000 \u0000 <p>The case involved a 51-year-old female patient with MOGAD who showed hypersomnolence. She had been diagnosed with MOGAD at 50 years old and admitted due to the development of optic neuritis and encephalitis while under corticosteroid and immunosuppressant treatment. These were improved after the introduction of intravenous methylprednisolone and plasma exchange. The orexin level in the cerebrospinal fluid (CSF) was decreased to 142.2 pg/mL (<200 pg/mL) upon admission and improved to a normal level (298.3 pg/mL) after immunotherapy. Bilateral hypothalamic lesions and reduction of the orexin level in the CSF were considered associated with hypersomnia.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Conclusion</h3>\u0000 \u0000 <p>The orexin level in CSF was useful to monitor hypothalamic dysfunction in a patient with MOGAD.</p>\u0000 </section>\u0000 </div>","PeriodicalId":10193,"journal":{"name":"Clinical and Experimental Neuroimmunology","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2022-08-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"44867742","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
A 36-year-old women developed dysarthria, dizziness, right-sided weakness, bilateral clumsiness, unsteady gait and fever (37.3 (cid:1) C) 1 day after a cold. The symptoms were aggravated 5 days later. The right limb was completely immobile. She could not understand the speech of others and could not be understood by others. Two days after the aggravation, high-dose methylprednisolone pulse therapy was given (1000 mg/day for 4 days, then 500 mg/day for 2 days), but her condi-tion continued to deteriorate. The patient was admitted to our hospital 14 days after the onset of the disease. On admission, the right muscle strength was grade 0, with low muscular tone and enhanced deep tendon reflexes. The right patellar clonus, ankle clonus, the Babinski sign and the Chaddock sign were positive. Magnetic resonance imaging (MRI) of the brain showed multiple lesions in bilateral frontal lobes, parietal lobes, lateral ventricle, basal ganglia regions, hippocampus, left thalamus, left occipital lobe, right cerebellar hemisphere, left brainstem and splenium of the corpus cal-losum. Unclassical concentric circle-like bands on T1- and T2-weighted images, fluid-attenuated inversion diffusion-weighted No in the post-contrast T1-weighted
{"title":"Balo's concentric sclerosis or multiple sclerosis? A demyelinating disease in a 36-year-old woman","authors":"Jingya Wei, Sa Wang, Juan Kang","doi":"10.1111/cen3.12727","DOIUrl":"10.1111/cen3.12727","url":null,"abstract":"A 36-year-old women developed dysarthria, dizziness, right-sided weakness, bilateral clumsiness, unsteady gait and fever (37.3 (cid:1) C) 1 day after a cold. The symptoms were aggravated 5 days later. The right limb was completely immobile. She could not understand the speech of others and could not be understood by others. Two days after the aggravation, high-dose methylprednisolone pulse therapy was given (1000 mg/day for 4 days, then 500 mg/day for 2 days), but her condi-tion continued to deteriorate. The patient was admitted to our hospital 14 days after the onset of the disease. On admission, the right muscle strength was grade 0, with low muscular tone and enhanced deep tendon reflexes. The right patellar clonus, ankle clonus, the Babinski sign and the Chaddock sign were positive. Magnetic resonance imaging (MRI) of the brain showed multiple lesions in bilateral frontal lobes, parietal lobes, lateral ventricle, basal ganglia regions, hippocampus, left thalamus, left occipital lobe, right cerebellar hemisphere, left brainstem and splenium of the corpus cal-losum. Unclassical concentric circle-like bands on T1- and T2-weighted images, fluid-attenuated inversion diffusion-weighted No in the post-contrast T1-weighted","PeriodicalId":10193,"journal":{"name":"Clinical and Experimental Neuroimmunology","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2022-08-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"46460273","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
In recent years, remarkable advances have been made in the treatment of multiple sclerosis (MS) and neuromyelitis optica spectrum disorder (NMOSD). In particular, several monoclonal antibody drugs have been approved for the treatment of MS and NMOSD, significantly expanding the therapeutic options for treating these diseases. This article provides an overview of the monoclonal antibodies available to treat patients with MS and NMOSD, including drugs that are not yet approved in Japan.
{"title":"Monoclonal antibody drugs for the treatment of multiple sclerosis and neuromyelitis optica spectrum disorder","authors":"Hisashi Murata, Yasuko Sugiyama, Naoyuki Shiraishi, Keigo Kihara, Makoto Kinoshita, Tatsusada Okuno","doi":"10.1111/cen3.12725","DOIUrl":"10.1111/cen3.12725","url":null,"abstract":"<p>In recent years, remarkable advances have been made in the treatment of multiple sclerosis (MS) and neuromyelitis optica spectrum disorder (NMOSD). In particular, several monoclonal antibody drugs have been approved for the treatment of MS and NMOSD, significantly expanding the therapeutic options for treating these diseases. This article provides an overview of the monoclonal antibodies available to treat patients with MS and NMOSD, including drugs that are not yet approved in Japan.</p>","PeriodicalId":10193,"journal":{"name":"Clinical and Experimental Neuroimmunology","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2022-07-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"41409283","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Multiple sclerosis (MS) and neuromyelitis optica (NMOSD) are more prevalent in women and mainly affecting young women, the majority of whom are of childbearing age. In addition, recent treatment algorithms suggest that patients who have poor prognostic factors are treated with highly effective disease-modifying drugs from the beginning. Monoclonal antibodies for MS or NMOSD are basically highly effective disease-modifying drugs. Therefore, young women with MS or NMOSD will have more opportunities to receive monoclonal antibody treatment than ever before. Currently, five monoclonal antibodies for MS or NMOSD are available in Japan: natalizumab and ofatumumab for MS, and eculizumab, satralizumab and inebilizumab for NMOSD. The pregnancy and breastfeeding of each monoclonal antibody drug is reviewed, and the evidence surrounding the safety of monoclonal antibody drugs during both pregnancy and breastfeeding in women with MS or NMOSD is discussed.
{"title":"Monoclonal antibody treatment during pregnancy and lactation in women with multiple sclerosis or neuromyelitis optica spectrum disorder","authors":"Chiyoko Nohara","doi":"10.1111/cen3.12724","DOIUrl":"10.1111/cen3.12724","url":null,"abstract":"<p>Multiple sclerosis (MS) and neuromyelitis optica (NMOSD) are more prevalent in women and mainly affecting young women, the majority of whom are of childbearing age. In addition, recent treatment algorithms suggest that patients who have poor prognostic factors are treated with highly effective disease-modifying drugs from the beginning. Monoclonal antibodies for MS or NMOSD are basically highly effective disease-modifying drugs. Therefore, young women with MS or NMOSD will have more opportunities to receive monoclonal antibody treatment than ever before. Currently, five monoclonal antibodies for MS or NMOSD are available in Japan: natalizumab and ofatumumab for MS, and eculizumab, satralizumab and inebilizumab for NMOSD. The pregnancy and breastfeeding of each monoclonal antibody drug is reviewed, and the evidence surrounding the safety of monoclonal antibody drugs during both pregnancy and breastfeeding in women with MS or NMOSD is discussed.</p>","PeriodicalId":10193,"journal":{"name":"Clinical and Experimental Neuroimmunology","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2022-07-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"41323332","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Shingo Konno, Takafumi Uchi, Jun Isonishi, Mari Matsushima, Hideo Kihara, Hideki Sugimoto, Toshiki Fujioka
Objectives
Evidence on factors contributing to the development of infections in myasthenia gravis (MG) patients on immunotherapy is scarce.
Methods
We studied 192 MG patients attending our hospital between April 2000 and May 2021. We examined the data of patients who had undergone immunotherapy and developed an infection and analyzed factors influencing infectious events including MG severity, antibody type, thymoma, thymectomy, treatment regimens and duration, and status of MG.
Results
A total of 148/192 (77%) patients (52 men, mean onset 43 y) underwent immunotherapy. Of these, 22/148 (14.8%) patients developed an infection-related hospitalization within 10 y of starting immunotherapy. Respiratory infections occurred in 14/22 (63.6%) of patients. The infections were fatal in 6/22 (27.2%) of patients. Infection-associated myasthenic crisis developed in 4/22 (18.1%) patients. Age at MG onset was the only variable associated with the development of infection (hazard ratio [HR]; 1.056, 95% confidence interval (95% CI): 1.0291.085, P < .001). The infection-free rate within 10 y of starting immunotherapy by MG subtype was 83.5% (95% CI: 61.4–93.5%) in ocular-MG (n = 29), and 87.5% (95% CI: 72.3–94.7%) in generalized early-onset MG (n = 55), 46.1% (95% CI: 21.7–67.6%) in generalized late-onset MG (n = 22), 87.7% (95% CI; 66.3–95.9%) in thymoma-associated MG (n = 29). Patients with muscle-specific tyrosine kinase antibody-positive MG (n = 2) and antibodies-negative MG (n = 11) did not experience infections.
Conclusion
Age at onset of MG was a significant contributor to the development of infection. Generalized late-onset MG is the most susceptible to infection and should be carefully monitored during immunotherapy.
关于重症肌无力（MG）患者在免疫治疗中导致感染发展的因素的证据很少。
{"title":"Development of infections among patients with myasthenia gravis undergoing immunotherapy","authors":"Shingo Konno, Takafumi Uchi, Jun Isonishi, Mari Matsushima, Hideo Kihara, Hideki Sugimoto, Toshiki Fujioka","doi":"10.1111/cen3.12723","DOIUrl":"10.1111/cen3.12723","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Objectives</h3>\u0000 \u0000 <p>Evidence on factors contributing to the development of infections in myasthenia gravis (MG) patients on immunotherapy is scarce.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>We studied 192 MG patients attending our hospital between April 2000 and May 2021. We examined the data of patients who had undergone immunotherapy and developed an infection and analyzed factors influencing infectious events including MG severity, antibody type, thymoma, thymectomy, treatment regimens and duration, and status of MG.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>A total of 148/192 (77%) patients (52 men, mean onset 43 y) underwent immunotherapy. Of these, 22/148 (14.8%) patients developed an infection-related hospitalization within 10 y of starting immunotherapy. Respiratory infections occurred in 14/22 (63.6%) of patients. The infections were fatal in 6/22 (27.2%) of patients. Infection-associated myasthenic crisis developed in 4/22 (18.1%) patients. Age at MG onset was the only variable associated with the development of infection (hazard ratio [HR]; 1.056, 95% confidence interval (95% CI): 1.0291.085, <i>P <</i> .001). The infection-free rate within 10 y of starting immunotherapy by MG subtype was 83.5% (95% CI: 61.4–93.5%) in ocular-MG (<i>n</i> = 29), and 87.5% (95% CI: 72.3–94.7%) in generalized early-onset MG (<i>n</i> = 55), 46.1% (95% CI: 21.7–67.6%) in generalized late-onset MG (<i>n</i> = 22), 87.7% (95% CI; 66.3–95.9%) in thymoma-associated MG (<i>n</i> = 29). Patients with muscle-specific tyrosine kinase antibody-positive MG (<i>n</i> = 2) and antibodies-negative MG (<i>n</i> = 11) did not experience infections.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Conclusion</h3>\u0000 \u0000 <p>Age at onset of MG was a significant contributor to the development of infection. Generalized late-onset MG is the most susceptible to infection and should be carefully monitored during immunotherapy.</p>\u0000 </section>\u0000 </div>","PeriodicalId":10193,"journal":{"name":"Clinical and Experimental Neuroimmunology","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2022-07-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"49276937","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Various effective monoclonal antibodies (mAbs) have been approved for both multiple sclerosis (MS) and anti-aquaporin-4-seropositive neuromyelitis optica spectrum disorders worldwide, including in Japan. As these newer mAbs have distinct modes of action that effectively suppress the recurrence of inflammation and slow disability progression, they can modulate and interfere with the protective immune response against pathogens, resulting in various infectious complications. Among various mAbs, natalizumab (NTZ) has the highest risk of causing progressive multifocal leukoencephalopathy (PML), a rare but fatal opportunistic brain infection caused by John Cunningham polyomavirus. Switching from NTZ to B-cell-depleting mAbs, such as ocrelizumab, is also a possible risk factor for PML development. Alemtuzumab carries the risk of reactivation of varicella-zoster virus (VZV); therefore, prophylactic acyclovir treatment is required. NTZ has also been associated with VZV reactivation. Eculizumab can cause severe meningococcal infection due to Neisseria meningitis, and vaccination prior to treatment induction is required. Attention to the reactivation of hepatitis B or Mycobacterium tuberculosis is also needed during mAb therapy. Additionally, in the era of severe acute respiratory syndrome coronavirus 2 infection (COVID-19), the risk for of developing severe COVID-19 may be associated with some mAbs, such as B-cell-depleting agents. Thorough understanding and mitigation strategies for infectious risks are essential.
{"title":"Infectious issues of therapeutic monoclonal antibodies in multiple sclerosis and neuromyelitis optica spectrum disorders","authors":"Asako Tagawa","doi":"10.1111/cen3.12721","DOIUrl":"10.1111/cen3.12721","url":null,"abstract":"<p>Various effective monoclonal antibodies (mAbs) have been approved for both multiple sclerosis (MS) and anti-aquaporin-4-seropositive neuromyelitis optica spectrum disorders worldwide, including in Japan. As these newer mAbs have distinct modes of action that effectively suppress the recurrence of inflammation and slow disability progression, they can modulate and interfere with the protective immune response against pathogens, resulting in various infectious complications. Among various mAbs, natalizumab (NTZ) has the highest risk of causing progressive multifocal leukoencephalopathy (PML), a rare but fatal opportunistic brain infection caused by John Cunningham polyomavirus. Switching from NTZ to B-cell-depleting mAbs, such as ocrelizumab, is also a possible risk factor for PML development. Alemtuzumab carries the risk of reactivation of varicella-zoster virus (VZV); therefore, prophylactic acyclovir treatment is required. NTZ has also been associated with VZV reactivation. Eculizumab can cause severe meningococcal infection due to <i>Neisseria meningitis</i>, and vaccination prior to treatment induction is required. Attention to the reactivation of hepatitis B or <i>Mycobacterium tuberculosis</i> is also needed during mAb therapy. Additionally, in the era of severe acute respiratory syndrome coronavirus 2 infection (COVID-19), the risk for of developing severe COVID-19 may be associated with some mAbs, such as B-cell-depleting agents. Thorough understanding and mitigation strategies for infectious risks are essential.</p>","PeriodicalId":10193,"journal":{"name":"Clinical and Experimental Neuroimmunology","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2022-07-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1111/cen3.12721","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"44933239","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Ana Rita Silva, Helena Felgueiras, Ana Isabel Gonçalves, Andreia Fernandes, Bruna Meira, Diana Melância, José Rosa, Maria Teresa Silvério, Ana Macedo
Objective
There is little research on the relationship between self-concept, psychiatric symptoms and quality of life among multiple sclerosis (MS) patients. We assessed the impact of disease perception (expectation and knowledge) on these metrics according to time from diagnosis.
Methods
This was an observational, cross-sectional, multicenter study. Group 1 included patients up to 3 months from MS diagnosis, whereas group 2 included patients with MS diagnosis established for >12 months and <36 months. A 19-item true/false questionnaire developed by the investigators to assess disease perception, Hospital Anxiety and Depression Scale (HADS), Fatigue Severity Scale and three-level level version of EQ-5D questionnaires were used.
Results
A total of 90 patients from six centers were included (38 in group 1). A total of 80% had a good disease knowledge, whereas just 48% reported positive expectations. There were no differences in disease knowledge, disease expectations, HADS, Fatigue Severity Scale and EQ-5D. We found an inverse correlation between disease knowledge and problems in self-care (P = 0.018) and fatigue (P = 0.032). Patients with the worst expectations about the disease were more anxious (P = 0.012 on HADS and P < 0.001 on EQ-5D). They also reported more problems in mobility (P = 0.002), self-care (P = 0.005), usual activities (p = 0.009) and pain (P = 0.001), and a worst health status compared with the past 12 months (P < 0.001) and with the best imaginable status (P < 0.001).
Conclusions
Our study showed no association between disease duration and disease perception. Patients with less disease knowledge reported more problems in self-care and higher fatigue scores. Patients with the worst disease expectations were more anxious and reported a worse health status. More attention should be paid to perceived health status in MS patients.
{"title":"Disease perception impacts quality of life and fatigue in relapsing–remitting multiple sclerosis patients","authors":"Ana Rita Silva, Helena Felgueiras, Ana Isabel Gonçalves, Andreia Fernandes, Bruna Meira, Diana Melância, José Rosa, Maria Teresa Silvério, Ana Macedo","doi":"10.1111/cen3.12720","DOIUrl":"10.1111/cen3.12720","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Objective</h3>\u0000 \u0000 <p>There is little research on the relationship between self-concept, psychiatric symptoms and quality of life among multiple sclerosis (MS) patients. We assessed the impact of disease perception (expectation and knowledge) on these metrics according to time from diagnosis.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>This was an observational, cross-sectional, multicenter study. Group 1 included patients up to 3 months from MS diagnosis, whereas group 2 included patients with MS diagnosis established for >12 months and <36 months. A 19-item true/false questionnaire developed by the investigators to assess disease perception, Hospital Anxiety and Depression Scale (HADS), Fatigue Severity Scale and three-level level version of EQ-5D questionnaires were used.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>A total of 90 patients from six centers were included (38 in group 1). A total of 80% had a good disease knowledge, whereas just 48% reported positive expectations. There were no differences in disease knowledge, disease expectations, HADS, Fatigue Severity Scale and EQ-5D. We found an inverse correlation between disease knowledge and problems in self-care (<i>P</i> = 0.018) and fatigue (<i>P</i> = 0.032). Patients with the worst expectations about the disease were more anxious (<i>P</i> = 0.012 on HADS and <i>P</i> < 0.001 on EQ-5D). They also reported more problems in mobility (<i>P</i> = 0.002), self-care (<i>P</i> = 0.005), usual activities (<i>p</i> = 0.009) and pain (<i>P</i> = 0.001), and a worst health status compared with the past 12 months (<i>P</i> < 0.001) and with the best imaginable status (<i>P</i> < 0.001).</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Conclusions</h3>\u0000 \u0000 <p>Our study showed no association between disease duration and disease perception. Patients with less disease knowledge reported more problems in self-care and higher fatigue scores. Patients with the worst disease expectations were more anxious and reported a worse health status. More attention should be paid to perceived health status in MS patients.</p>\u0000 </section>\u0000 </div>","PeriodicalId":10193,"journal":{"name":"Clinical and Experimental Neuroimmunology","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2022-07-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"48953080","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Postvaccination acute disseminated encephalomyelitis (ADEM) may develop 2–30 days after vaccination. However, ADEM following pneumococcal vaccination has never been reported.
Case presentation
We report a 73-year-old woman with ADEM following pneumococcal vaccination. She developed acute fever and consciousness disturbance 17 days after the second administration of 23-valent pneumococcal polysaccharide vaccine (PPSV23). Head magnetic resonance imaging (MRI) revealed numerous T2-high lesions, mainly involving the cerebral and cerebellar white matter and brainstem. Cerebrospinal fluid examination showed polymorphonuclear pleocytosis and markedly elevated interleukin-6 (58,400 pg/ml). Her symptoms and MRI lesions were promptly resolved by steroid pulse therapy, with no relapse.
Conclusion
Although PPSV23 is recommended for elderly individuals, this case highlights the risk of ADEM caused by unexpected immune activation following repeated administration of the vaccine.
{"title":"Acute disseminated encephalomyelitis in an elderly patient following pneumococcal vaccination with extremely high cerebrospinal fluid interleukin-6","authors":"Yuu-ichi Kira, Takumi Tashiro, Norihisa Maeda","doi":"10.1111/cen3.12719","DOIUrl":"10.1111/cen3.12719","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Background</h3>\u0000 \u0000 <p>Postvaccination acute disseminated encephalomyelitis (ADEM) may develop 2–30 days after vaccination. However, ADEM following pneumococcal vaccination has never been reported.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Case presentation</h3>\u0000 \u0000 <p>We report a 73-year-old woman with ADEM following pneumococcal vaccination. She developed acute fever and consciousness disturbance 17 days after the second administration of 23-valent pneumococcal polysaccharide vaccine (PPSV23). Head magnetic resonance imaging (MRI) revealed numerous T2-high lesions, mainly involving the cerebral and cerebellar white matter and brainstem. Cerebrospinal fluid examination showed polymorphonuclear pleocytosis and markedly elevated interleukin-6 (58,400 pg/ml). Her symptoms and MRI lesions were promptly resolved by steroid pulse therapy, with no relapse.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Conclusion</h3>\u0000 \u0000 <p>Although PPSV23 is recommended for elderly individuals, this case highlights the risk of ADEM caused by unexpected immune activation following repeated administration of the vaccine.</p>\u0000 </section>\u0000 </div>","PeriodicalId":10193,"journal":{"name":"Clinical and Experimental Neuroimmunology","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2022-07-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1111/cen3.12719","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"41528073","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
The launch of highly efficacious monoclonal antibody (mAb) drugs has profoundly changed the therapeutic strategy for multiple sclerosis and neuromyelitis optica spectrum disorders (NMOSD). However, the newly developed mAb drugs, especially those used in treating NMOSD, are quite expensive, resulting in a significant medical and economic burden. Early use of these drugs is expected to reduce the frequency of relapse and the accumulation of disability, preserve the quality of life of patients, and improve their life expectancy. In selecting therapeutic drugs, it is necessary not only to consider the efficacy and safety of the drugs, but also to consider the cost-effectiveness of each drug for each patient, considering the economic aspects of health care. However, the cost-effectiveness of mAb drugs in treating multiple sclerosis and NMOSD has not been fully verified. Japan is rapidly emerging as a hyper-aged society, unlike any other in the world, and the rising cost of medical care is an urgent issue that needs to be addressed and resolved. Out-of-pocket expenses of individual patients are not a major issue in Japan as medical, costs for multiple sclerosis and NMOSD are subsidized by public medical insurance; however, the judicious use of mAb drugs is required to ensure cost-effectiveness.
{"title":"The impact of monoclonal antibody drugs on healthcare economics in the treatment of multiple sclerosis and neuromyelitis optica spectrum disorders","authors":"Takashi Ohashi","doi":"10.1111/cen3.12718","DOIUrl":"10.1111/cen3.12718","url":null,"abstract":"<p>The launch of highly efficacious monoclonal antibody (mAb) drugs has profoundly changed the therapeutic strategy for multiple sclerosis and neuromyelitis optica spectrum disorders (NMOSD). However, the newly developed mAb drugs, especially those used in treating NMOSD, are quite expensive, resulting in a significant medical and economic burden. Early use of these drugs is expected to reduce the frequency of relapse and the accumulation of disability, preserve the quality of life of patients, and improve their life expectancy. In selecting therapeutic drugs, it is necessary not only to consider the efficacy and safety of the drugs, but also to consider the cost-effectiveness of each drug for each patient, considering the economic aspects of health care. However, the cost-effectiveness of mAb drugs in treating multiple sclerosis and NMOSD has not been fully verified. Japan is rapidly emerging as a hyper-aged society, unlike any other in the world, and the rising cost of medical care is an urgent issue that needs to be addressed and resolved. Out-of-pocket expenses of individual patients are not a major issue in Japan as medical, costs for multiple sclerosis and NMOSD are subsidized by public medical insurance; however, the judicious use of mAb drugs is required to ensure cost-effectiveness.</p>","PeriodicalId":10193,"journal":{"name":"Clinical and Experimental Neuroimmunology","volume":null,"pages":null},"PeriodicalIF":0.0,"publicationDate":"2022-07-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"41531402","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Christopher Drudge, Melody Zhao, Satoru Tanaka, Nozomu Tanaka, Hiromichi Otaka, Izumi Kawachi, Dieter A. Häring, Róisín Brennan, Nicholas Adlard, Imtiaz A. Samjoo
Objective
No head-to-head clinical trials have compared ofatumumab with other disease-modifying therapies (DMTs) available in Japan for patients with relapsing multiple sclerosis (RMS). In this study, a network meta-analysis (NMA) was conducted to compare the efficacy of ofatumumab to other DMTs currently available in Japan for the treatment of patients with RMS.
Methods
Systematic searches were conducted in biomedical databases from inception to June 2020 to identify randomized controlled trials. Only English- and Japanese-language publications describing studies conducted in Japan were included. Trials with sufficiently similar study and patient characteristics were included in a Bayesian NMA. A sensitivity analysis was conducted to explore the impact of potential sources of uncertainty.
Results
Four trials, each comparing a DMT with placebo in a ≥50% Japanese population, were sufficiently similar that comparative efficacy could be assessed for annualized relapse rate (ARR). Ofatumumab numerically reduced ARR compared with fingolimod (rate ratio [RR]: 0.84, 95% credible interval [CrI]: 0.20–3.39), dimethyl fumarate (RR: 0.61, 95% CrI: 0.16–2.30), and placebo (RR: 0.41, 95% CrI: 0.12–1.39), but not natalizumab (RR: 1.33, 95% CrI: 0.33–5.45). In a subgroup analysis of Japanese patients only, ofatumumab reduced relapses compared with all other treatments including natalizumab. These results were limited by the lack of studies reporting direct comparisons between included treatments and by heterogenous reporting of outcome data.
Conclusion
These findings, although limited by the paucity of evidence for Japanese patients, suggest that monoclonal antibody therapies (ie, natalizumab and ofatumumab) may provide improved efficacy compared with other DMTs available in Japan for patients with RMS.
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