Association and linkage of juvenile MS with HLA-DR2(15) in Russians

Бойко, А. Н.; Gusev, E I; Судомоина, М. А.; Alekseenkov, A. D.; Кулакова, О. Г.; Bikova, O. V.; Маслова, О. И.; Boiko, Svetlana; Гусева, М Е; Фаворова, О. О.

doi:10.1212/wnl.58.4.658

Cited by 52 publications

(37 citation statements)

References 7 publications

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“…102 Similar overall findings have been reported for pediatric MS in two studies from Russia. 112,113 Similar frequencies of DRB1 alleles and genotypes were identified, regardless of whether the onset of MS was younger than 16 years of age (n ϭ 56) or 16 and older (n ϭ 234). 112 The other study of a Russian cohort suggested an association between DR15 (DRB1*150) alleles and susceptibility in children to ON and MS. 113 Conclusion.…”

Section: Disease Course and Outcomementioning

confidence: 82%

“…112,113 Similar frequencies of DRB1 alleles and genotypes were identified, regardless of whether the onset of MS was younger than 16 years of age (n ϭ 56) or 16 and older (n ϭ 234). 112 The other study of a Russian cohort suggested an association between DR15 (DRB1*150) alleles and susceptibility in children to ON and MS. 113 Conclusion. In the initial stages, pediatric MS is associated with a more favorable outcome compared to adult MS; however, over the long term, pediatric MS patients can become disabled at a younger age, underscoring that MS in pediatric population does not have a benign prognosis and early initiation of DMT should be considered.…”

Section: Disease Course and Outcomementioning

confidence: 82%

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Clinical features of children and adolescents with multiple sclerosis

Ness

Chabas²,

Sadovnick³

et al. 2007

Neurology

108

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There is increasing appreciation that multiple sclerosis (MS) can begin in childhood or adolescence, but pediatric MS continues to be a rare entity, with an estimated 2 to 5% of patients with MS experiencing their first clinical symptoms before age 16. A prompt diagnosis of pediatric MS is important to optimize overall management of both the physical and social impact of the disease. The widespread use of disease-modifying therapies (DMT) for MS in adults, as early as following an initial isolated episode, has led to the use of DMT in children and adolescents with MS. However, it is imperative to distinguish pediatric MS from other childhood CNS inflammatory demyelinating disorders such as acute disseminated encephalomyelitis. Although increasing evidence suggests a slower disease course in children with MS compared to adults, significant disability can still accumulate by early adulthood. Furthermore, associated neurocognitive deficits can impair both academic and psychosocial function at a critical juncture in a young person's life. This article reviews the clinical characteristics, neuroimaging, paraclinical findings, disease course, epidemiology, genetics, and pathophysiology of pediatric MS vis-à-vis adult MS. Further research of pediatric MS may advance our understanding of MS pathophysiology in general, as well as improve the long-term health care outcomes of children and adolescents diagnosed with MS.

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Section: Disease Course and Outcomementioning

confidence: 82%

Section: Disease Course and Outcomementioning

confidence: 82%

Clinical features of children and adolescents with multiple sclerosis

Ness

Chabas²,

Sadovnick³

et al. 2007

Neurology

108

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“…16,17 Bu konuda yapılmış en kapsamlı araştırmalardan biri Nielsen ve ark.nın çalışması olup, MS hastalarının birinci derece yakınlarında yedi kat risk artışı tanımlanmıştır. 18 Kuveyt'te yapılmış bir çalışmada ise aile öyküsü pediatrik baş-langıçlı hastalarda %12,6 erişkin başlangıçlı hastalarda %15,2 olarak bulunmuştur.…”

Section: Discussionunclassified

A Comparison of Pediatric- and Adult-Onset Multiple Sclerosis

Kamaşak¹,

Cansu²,

Arslan³

et al. 2017

Turkiye Klinikleri J Neur

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ultipl skleroz (MS); santral sinir sisteminin kronik, inflamatuar, demiyelinizan hastalığıdır. Sıklıkla 20'li yaşlardan sonra ortaya çıkmaktadır. MS, için pediatrik yaşlarda başlangıç çok sık değildir. Pediatrik yaş başlangıçlı MS, tüm MS hastalarının yaklaşık %2-10'unu kapsamaktadır.1 Geçtiğimiz yıllarda, "International Pediatric MS Study Group" (416) olarak gruplandırıldı. Her iki grup yaş, cinsiyet, hastalık süresi, sekonder relaps, başlangıç tutulumları, EDSS 6'ya ulaşma süreleri açısından karşılaştırıldı. B Bu ul lg gu ul la ar r: : Her iki grupta da kadın cinsiyet baskınlığı dikkat çekiciydi (2:1). Hastalık süreleri açısından değerlendirildiğinde, pediatrik yaş başlangıçlı MS hastaları daha uzun süredir MS tanısı ile izleniyorlardı. Supratentoryal tutulum erişkin ve pediatrik hastalarda en sık başlangıç kliniğine neden olan tutulum bölgesi idi. Sekonder relaps süreleri açısından gruplar arasında fark izlenmedi. EDSS 6'ya pediatrik başlangıçlı MS hastalarının daha uzun süre sonra ulaştığı gözlendi. S So on nu uç ç: : Çalışmamızda, 208 pediatrik başlangıçlı MS hastasının klinik özellikleri, erişkin başlangıçlı MS hastaları ile karşılaştırıldı. Pediatrik başlangıçlı MS hastalarının EDSS 6 skoruna daha uzun sürede ulaştığı görüldü. (208) and those diagnosed over the age of 18 as adult-onset MS (416). The two groups were compared in terms of age, sex, duration of disease, secondary relapse, onset involvements and time to Expanded Disability Status Scale (EDSS) 6. R Re es su ul lt ts s: : Predominance of female gender (2:1) was marked in both groups. Duration of disease was greater in patients with pediatric-onset MS. Supratentorial involvement was the most common cause of the initial clinical manifestation in both adult and pediatric patients. No difference was determined between the groups in terms of secondary relapse times. Time to EDSS 6 was longer in patients with pediatric-onset MS. C Co on nc cl lu us si io on n: : Our study compared the clinical characteristics of adult-onset MS with those of 208 pediatric-onset MS patients. Time to an EDSS score of 6 was longer in patients with pediatric-onset MS.K Ke ey yw wo or rd ds s:

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“…31 Several studies have shown a significant skew to HLA DRB1*1501 in pediatric patients with MS vs healthy controls or individuals with monophasic ADS. 4,32,33 A recent study in white pediatric patients with MS has confirmed that, in addition to the strong effect of DRB1*1501, 36 of the 110 genetic variants associated with adult MS are also associated with the disease in children. 34 In fact, a larger effect size was reported for several of these variants compared with adult MS, suggesting that genes may have a stronger effect on pediatric MS susceptibility.…”

Section: Genetic Risk Factors For Pediatric Ms Susceptibility and Dismentioning

confidence: 94%

Environmental and genetic factors in pediatric inflammatory demyelinating diseases

et al. 2016

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The onset of multiple sclerosis (MS) occurs in childhood in about 5% of all patients with MS. The disease in adults has a complex genetic and environmental inheritability. One of the main risk factors, also confirmed in pediatric MS, is HLA DRB1*1501. In addition to genetic factors, a large part of disease susceptibility in adults is conferred by environmental risk factors such as low vitamin D status, exposure to cigarette smoking, and remote Epstein-Barr virus (EBV) infection. In children, both exposure to cigarette smoking and prior EBV infection have been reported consistently as risk factors for MS. The role of vitamin D remains to be confirmed in this age category. Finally, although very likely critical in disease processes, few gene-environment interactions and epigenetic changes have been reported for adult and pediatric MS susceptibility. Of interest, some of the risk factors for MS have also been associated with disease course modification, such as low 25(OH) vitamin D serum levels in pediatric and adult MS. Age is also a clear disease modifier of clinical, CSF, and MRI phenotype in children with the disease. Finally, although much has yet to be unraveled regarding molecular processes at play in MS, there is a larger gap in our knowledge of genetic and environmental risk factors for pediatric neuromyelitis optica spectrum disorders and acute disseminated encephalomyelitis and only collaborative studies will answer those questions. Among pediatric acquired demyelinating disorders of the CNS, multiple sclerosis (MS), including clinically isolated syndromes (CIS), acute disseminated encephalomyelitis, and neuromyelitis optica (NMO) spectrum disorders are the most common. Due to its chronicity and proportionally higher incidence, substantial efforts have focused on understanding factors contributing to MS, although the study of risk factors for NMO is also accelerating. 1Despite the fact that several genetic and environmental risk factors have been consistently reported for adult MS, 2 underlying molecular processes at play remain largely unknown. The onset of MS in children, although representing only approximately 5% of all MS onsets, leads us to question whether the load of genetic factors and environmental exposures contributing to disease onset may in fact be higher than in adults who develop the disease. As such environmental risk factors should be easier to identify in younger patients and if disease processes at play overlap in all age groups, this information might be extended to adult-onset MS. It is unknown why disease onset is relatively rare in children vs adults. This could be in part due to developmental changes in the CNS or immune systems.Exposures to environmental risk factors for adult MS are believed to occur for the most part before age 15.2 As such, studying risk factors in pediatric MS during the putative window of disease susceptibility may considerably shorten the time between exposure action and time of

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Association and linkage of juvenile MS with HLA-DR2(15) in Russians

Cited by 52 publications

References 7 publications

Clinical features of children and adolescents with multiple sclerosis

Clinical features of children and adolescents with multiple sclerosis

A Comparison of Pediatric- and Adult-Onset Multiple Sclerosis

Environmental and genetic factors in pediatric inflammatory demyelinating diseases

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