mrjСовременная ревматологияModern Rheumatology Journal1996-70122310-158XIMA-PRESS, LLC10.14412/1996-7012-2022-4-32-39mrj-1320Research ArticleОРИГИНАЛЬНЫЕ ИССЛЕДОВАНИЯORIGINAL INVESTIGATIONSЮвенильные идиопатические воспалительные миопатии: результаты открытого одноцентрового ретроспективного исследованияJuvenile idiopathic inflammatory myopathies: results of an open single-center retrospective studyhttps://orcid.org/0000-0002-0513-6826КаледаМ. И.KaledaM. I.

 Мария Игоревна Каледа

 Россия, 115522, Москва, Каширское шоссе, 34А 

 Maria Igorevna Kaleda 

 34A, Kashirskoe shosse, Moscow 115522, Russia 

kaleda-mi@yandex.ruhttps://orcid.org/0000-0003-1842-0348НикишинаИ. П.NikishinaI. P.

 Россия, 115522, Москва, Каширское шоссе, 34А 

 34A, Kashirskoe shosse, Moscow 115522, Russia 

https://orcid.org/0000-0003-3689-431XСалугинаС. О.SaluginaS. O.

 Россия, 115522, Москва, Каширское шоссе, 34А 

 34A, Kashirskoe shosse, Moscow 115522, Russia 

https://orcid.org/0000-0003-2671-1655ФедоровЕ. С.FedorovE. S.

 Россия, 115522, Москва, Каширское шоссе, 34А 

 34A, Kashirskoe shosse, Moscow 115522, Russia 

https://orcid.org/0000-0001-9803-0221АрсеньеваС. В.ArsenyevaS. V.

 Россия, 115522, Москва, Каширское шоссе, 34А 

 34A, Kashirskoe shosse, Moscow 115522, Russia 

https://orcid.org/0000-0003-1648-7848ШаповаленкоА. Н.ShapovalenkoA. N.

 Россия, 115522, Москва, Каширское шоссе, 34А 

 34A, Kashirskoe shosse, Moscow 115522, Russia 

https://orcid.org/0000-0001-6183-8630ПачкорияТ. Н.PachkoriaT. N.

 Россия, 115522, Москва, Каширское шоссе, 34А 

 34A, Kashirskoe shosse, Moscow 115522, Russia 

https://orcid.org/0000-0002-3815-0608МаткаваВ. Г.MatkavaV. G.

 Россия, 115522, Москва, Каширское шоссе, 34А 

 34A, Kashirskoe shosse, Moscow 115522, Russia 

ФГБНУ «Научно-исследовательский институт ревматологии им. В.А. Насоновой»РоссияV.A. Nasonova Research Institute of RheumatologyRussian Federation2022200820221643239Copyright © Каледа М.И., Никишина И.П., Салугина С.О., Федоров Е.С., Арсеньева С.В., Шаповаленко А.Н., Пачкория Т.Н., Маткава В.Г., 20222022Каледа М.И., Никишина И.П., Салугина С.О., Федоров Е.С., Арсеньева С.В., Шаповаленко А.Н., Пачкория Т.Н., Маткава В.Г.Kaleda M.I., Nikishina I.P., Salugina S.O., Fedorov E.S., Arsenyeva S.V., Shapovalenko A.N., Pachkoria T.N., Matkava V.G.Данная работа распространяется под лицензией Creative Commons Attribution 4.0.This work is licensed under a Creative Commons Attribution 4.0 License.https://mrj.ima-press.net/mrj/article/view/1320

Ювенильные идиопатические воспалительные миопатии (ЮИВМ) – редкие заболевания, при которых прогноз во многом определяется своевременной диагностикой, сроками назначения и эффективностью терапии.Цель исследования – охарактеризовать клинические фенотипы, результаты параклинических методов обследования, спектр аутоантител, а также терапевтические возможности у пациентов с ЮИВМ.Пациенты и методы. В ретроспективное исследование включено 37 пациентов с ЮИВМ, госпитализированных в детское отделение ФГБНУ «Научно-исследовательский институт ревматологии им. В.А. Насоновой» с 2016 по 2020 г. Всем больным проводилось стандартное клиническое и лабораторно-инструментальное обследование в соответствии с диагнозом и тяжестью состояния.Результаты и обсуждение. У 23 из 37 пациентов с ЮИВМ имелся ювенильный дерматомиозит (ЮДМ), у 1 – полимиозит и у 13 – overlap-миозит (ОМ). Соотношение мальчиков и девочек – 1:1,7. Медиана возраста дебюта ЮДМ составляла 6,9 года, ОМ – 11,3 года. У всех пациентов отмечалось поражение скелетной мускулатуры, дисфагия выявлена в 52,2% случаях ЮДМ и в 15,4% случаев ОМ. Повышение уровня креатинфосфокиназы на момент установления диагноза наблюдалось у 72,9% пациентов, лактатдегидрогеназы – у 81,1%, аланинаминотрансферазы – у 67,6%, аспартатаминотрансферазы – у 75,7%. Гелиотропная сыпь и/или симптом Готтрона отмечались у 100% пациентов с ЮДМ и у 30,8% с ОМ. Поражение легких выявлено у 27% детей. Миопатический тип нарушений при капилляроскопии имелся у 95,2% пациентов с ЮДМ и 53,8% с ОМ. У 10,8% пациентов с ЮИВМ были обнаружены миозит-специфические аутоантитела. Все пациенты получали глюкокортикоиды, 81,0% – метотрексат, 18,9% – гидроксихлорохин, 8,1% – циклофосфамид, 8,1% – циклоспорин, 2,7% – микофенолата мофетил, 2,7% – азатиоприн, 67,6% – внутривенный иммуноглобулин. Генно-инженерные биологические препараты (ГИБП) были назначены 26% пациентов с ЮДМ и всем больным с ОМ (всего в 51,3% случаев при ЮИВМ). Медиана продолжительности заболевания до инициации терапии ГИБП составляла 2,25 года. 58,8% пациентов проводилась терапия ритуксимабом (РТМ), 41,2% – абатацептом (АБЦ). У 1 пациента с ОМ, представленным сочетанием системной красной волчанки и ЮДМ, последовательно применялись три ГИБП: АБЦ, этанерцепт и РТМ. Все пациенты достигли неактивного статуса болезни.Заключение. ЮДМ – наиболее частый фенотип ЮИВМ, для которого характерны более ранний возраст дебюта, появление кожного синдрома, предшествующее развитию миопатии, типичные капилляроскопические изменения. Сделан вывод о высокой эффективности и приемлемом профиле безопасности терапии ГИБП у детей с ЮИВМ при условии тщательного мониторинга ее переносимости. ГИБП могут назначаться уже на ранних сроках заболевания при наличии прогностически неблагоприятных факторов.

Juvenile idiopathic inflammatory myopathies (JIIM) are rare diseases in which the prognosis is largely determined by timely diagnosis, timing of prescription and effectiveness of therapy.Objective: to characterize the clinical phenotypes, the results of paraclinical examination methods, the spectrum of autoantibodies, as well as therapeutic options in patients with JIIM.Patients and methods. The retrospective study included 37 patients with JIIM hospitalized in the pediatric department of the V.A. Nasonova Research Institute of Rheumatology from 2016 to 2020. All patients underwent a standard clinical and laboratory-instrumental examination in accordance with the diagnosis and severity of the condition.Results and discussion. Twenty-three of the 37 JIIM patients had juvenile dermatomyositis (JDM), 1 had polymyositis, and 13 had overlap-myositis (OM). The ratio of boys and girls was 1:1.7. The median age of onset for JDM was 6.9 years, and OM was 11.3 years. All patients had skeletal muscles involvement, dysphagia was detected in 52.2% of cases of JDM and in 15.4% of cases of OM. An increase in the level of creatine phosphokinase at the time of diagnosis was observed in 72.9% of patients, of lactate dehydrogenase – in 81.1%, of alanine aminotransferase - in 67.6%, of aspartate aminotransferase – in 75.7%. Heliotrope rash and/or Gottron's syndrome were observed in 100% of patients with JDM and in 30.8% with OM. Lung involvement was found in 27% of children. The myopathic capillaroscopic changes were present in 95.2% of patients with JDM and 53.8% with OM. Myositis-specific autoantibodies were found in 10.8% of patients with JIIM.All patients received glucocorticoids, 81.0% methotrexate, 18.9% hydroxychloroquine, 8.1% cyclophosphamide, 8.1% cyclosporine, 2.7% mycophenolate mofetil, 2.7% azathioprine, 67.6% - intravenous immunoglobulin. Biologic disease modifying antirheumatic drugs (bDMARDs) were prescribed to 26% of patients with JDM and to all patients with OM (only in 51.3% of cases with JIIM). The median duration of illness before initiation of bDMARDs therapy was 2.25 years. 58.8% of patients were treated with rituximab (RTM), 41.2% with abatacept (ABA). In 1 patient with OM, represented by a combination of systemic lupus erythematosus and JDM, three bDMARDs were used sequentially: ABA, etanercept and RTM. All patients achieved inactive disease status.Conclusion. JDM is the most common phenotype of JIIM, which is characterized by an earlier age of onset, skin involvement that precedes the development of myopathy, and typical capillaroscopic changes. The conclusion was made about the high efficacy and acceptable safety profile of bDMARDs therapy in children with JIIM, with careful monitoring of its tolerability. bDMARDs can be prescribed even in the early stages of the disease in the presence of unfavorable prognostic factors.

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The authors declare that there are no conflicts of interest present.