akusherstvoObstetrics, Gynecology and ReproductionАкушерство, Гинекология и Репродукция2313-73472500-3194IRBIS LLC10.17749/2313-7347/ob.gyn.rep.2024.498akusherstvo-2291Research ArticleCLINICAL CASESКЛИНИЧЕСКИЕ СЛУЧАИMacrophage activation syndrome during pregnancy as a fatal complication of systemic idiopathic arthritisСиндром активации макрофагов во время беременности как фатальное осложнение системного идиопатического артритаhttps://orcid.org/0000-0001-8652-1410ШестерняП. А.ShesternyaP. A.

Шестерня Павел Анатольевич, д.м.н., проф.

660022 Красноярск, ул. Партизана Железняка, д. 1

WoS ResearcherID: M-3898-2014

Pavel A. Shesternya. MD, Dr Sci Med, Prof. 

WoS ResearcherID: M-3898-2014

1 Partizana Zheleznyaka Str., Krasnoyarsk 660022

https://orcid.org/0000-0003-2228-3884ЦхайВ. Б.TskhayV. B.

Цхай Виталий Борисович, д.м.н., проф.

660022 Красноярск, ул. Партизана Железняка, д. 1

WoS ResearcherID: C-4869-2014

Vitaly B. Tskhay, MD, Dr Sci Med, Prof. 

WoS ResearcherID: C-4869-2014

1 Partizana Zheleznyaka Str., Krasnoyarsk 660022

tchai@yandex.ruhttps://orcid.org/0000-0002-1971-632XБазинаМ. И.BazinaM. I.

Базина Марина Ивановна, д.м.н.

660022 Красноярск, ул. Партизана Железняка, д. 1

Marina I. Bazina, MD, Dr Sci Med.

1 Partizana Zheleznyaka Str., Krasnoyarsk 660022

МатвееваИ. В.MatveevaI. V.

Матвеева Ирина Владимировна

660022 Красноярск, ул. Партизана Железняка, д. 3

Irina V. Matveeva, MD.

3 Partizana Zheleznyaka Str., Krasnoyarsk 660022

ФГБОУ ВО «Красноярский государственный медицинский университет имени профессора В.Ф. Войно-Ясенецкого» Министерства здравоохранения Российской ФедерацииРоссияProfessor V.F. Voino-Yasenetsky Krasnoyarsk State Medical University, Health Ministry of Russian FederationRussian FederationКГБУЗ «Краевая клиническая больница»РоссияKrasnoyarsk Regional Clinical HospitalRussian Federation202410012025186898907Copyright © Shesternya P.A., Tskhay V.B., Bazina M.I., Matveeva I.V., 20252025Шестерня П.А., Цхай В.Б., Базина М.И., Матвеева И.В.Shesternya P.A., Tskhay V.B., Bazina M.I., Matveeva I.V.Данная работа распространяется под лицензией Creative Commons Attribution 4.0.This work is licensed under a Creative Commons Attribution 4.0 License.https://www.gynecology.su/jour/article/view/2291

According to the current concept, juvenile arthritis with a systemic onset (systemic juvenile idiopathic arthritis, sJIA) and adult­onset Still's disease (ASD) are considered as rare autoinflammatory diseases, with the underlying immunopathogenesis being based on the mechanisms bridging both of such diseases. Despite they develop in childhood and young age, the issues of reproduction occurring during these pathologies virtually unexplored. The article presents a clinical case of maternal death in a 22-year-old primiparous woman and analyzes the tactics of sJIA management, which was complicated by pregnancy-related macrophage activation syndrome.

Согласно современной концепции, юношеский артрит с системным началом (системный ювенильный идиопатический артрит, сЮИА) и болезнь Стилла взрослых (БСВ) рассматриваются как редкие аутовоспалительные заболевания, в имму­нопатогенезе которых лежат объединяющие оба данных заболевания механизмы. Несмотря на развитие заболеваний в детском и молодом возрасте, вопросы репродукции при этих заболеваниях остаются практически не изученными. В статье представлен клинический случай материнской смертности 22-летней первородящей женщины, проведен анализ тактики ведения сЮИА, осложнившийся синдромом активации макрофагов во время беременности.

системный ювенильный идиопатический артритсЮИАсиндром активации макрофаговболезнь Стилла взрослыхбеременностьтоцилизумабматеринская смертностьsystemic juvenile idiopathic arthritissJIAmacrophage activation syndromeadult-onset Still's diseasepregnancytocilizumabmaternal mortalityReferencesStill G.F. On a form of chronic joint disease in children. Med Chir Trans. 1897;80:47-60.9.Still G.F. On a form of chronic joint disease in children. Med Chir Trans. 1897;80:47-60.9.Bywaters E.G. Still's disease in the adult. Ann Rheum Dis. 1971;30(2):121-33. https://doi.Org/10.1136/ard.30.2.121.Bywaters E.G. Still's disease in the adult. Ann Rheum Dis. 1971;30(2):121-33. https://doi.org/10.1136/ard.30.2.121.Насонов Е.Л., Файст Е. Болезнь Стилла взрослых: новые горизонты. Научно-практическая ревматология. 2021;59(6):645-65. https://doi.org/10.47360/1995-4484-2021-643-663.Nasonov E.L., Feist E. Adult Still's disease: New horizons. [Bolezn' Stilla vzroslyh: novye gorizonty]. Nauchcno-prakticheskaya pevmatologia. 2021;59(6):645-65. (In Russ.). https://doi.org/10.47360/1995-4484-2021-643-663.Efthimiou P., Kontzias A., Hur P. et al. Adult-onset Still's disease in focus: Clinical manifestations, diagnosis, treatment, and unmet needs in the era of targeted therapies. Semin Arthritis Rheum. 2021;51(4):858-74. https://doi.org/10.1016/j.semarthrit.2021.06.004.Efthimiou P., Kontzias A., Hur P. et al. Adult-onset Still's disease in focus: Clinical manifestations, diagnosis, treatment, and unmet needs in the era of targeted therapies. Semin Arthritis Rheum. 2021;51(4):858-74. https://doi.org/10.1016/j.semarthrit.2021.06.004.Gerfaud-Valentin M., Hot A., Huissoud C. et al. Adult-onset Still disease and pregnancy: about ten cases and review of the literature. Rheumatol Int. 2014;34(6):867-71. https://doi.org/10.1007/s00296-013-2765-5.Gerfaud-Valentin M., Hot A., Huissoud C. et al. Adult-onset Still disease and pregnancy: about ten cases and review of the literature. Rheumatol Int. 2014;34(6):867-71. https://doi.org/10.1007/s00296-013-2765-5.De Carolis S., Cianci F., Del Sordo G. et al. Adult onset Still' and pregnancy. Autoimmun Rev. 2019;18(9):102356. https://doi.org/10.1016/j.autrev.2019.102356.De Carolis S., Cianci F., Del Sordo G. et al. Adult onset Still' and pregnancy. Autoimmun Rev. 2019;18(9):102356. https://doi.org/10.1016/j.autrev.2019.102356.Abdulkarim S., Otieno F., Ali S.K. Adult-onset Still's disease triggered by pregnancy. Proc (Bayl Univ Med Cent). 2019;32(2):229-30. https://doi.org/10.1080/08998280.2018.1562292.Abdulkarim S., Otieno F., Ali S.K. Adult-onset Still's disease triggered by pregnancy. Proc (Bayl Univ Med Cent). 2019;32(2):229-30. https://doi.org/10.1080/08998280.2018.1562292.Plapais L., Mekinian A., Bornes M. et al. Adult onset Still's disease occurring during pregnancy: Case-report and literature review. Semin Arthritis Rheum. 2018;47(4):575-7. https://doi.org/10.1016/j.semarthrit.2017.07.003.Plapais L., Mekinian A., Bornes M. et al. Adult onset Still's disease occurring during pregnancy: Case-report and literature review. Semin Arthritis Rheum. 2018;47(4):575-7. https://doi.org/10.1016/j.semarthrit.2017.07.003.Ursin K., Lydersen S., Skomsvoll J.F., Wallenius M. Disease juvenile idiopathic arthritis during and after pregnancy: A prospective multicenter study. J Rheumatol. 2018;45(2):257-65. https://doi.org/10.3899/jrheum.161410.Ursin K., Lydersen S., Skomsvoll J.F., Wallenius M. Disease juvenile idiopathic arthritis during and after pregnancy: A prospective multicenter study. J Rheumatol. 2018;45(2):257-65. https://doi.org/10.3899/jrheum.161410.Garda-Fernandez A., Gerardi M.C., Crisafulli F. et al. Disease course and obstetric outcomes of pregnancies in juvenile idiopathic arthritis: are there any differences among disease? A single-center retrospective study of prospectively followed pregnancies in a dedicated pregnancy clinic. Clin Rheumatol. 2021;40(1):239-44. https://doi.org/10.1007/s10067-020-05404-w.Garda-Fernandez A., Gerardi M.C., Crisafulli F. et al. Disease course and obstetric outcomes of pregnancies in juvenile idiopathic arthritis: are there any differences among disease? A single-center retrospective study of prospectively followed pregnancies in a dedicated pregnancy clinic. Clin Rheumatol. 2021;40(1):239-44. https://doi.org/10.1007/s10067-020-05404-w.Lenert A., Yao Q. Macrophage activation syndrome complicating adult onset Still's disease: A single center case series and comparison with literature. Semin Arthritis Rheum. 2016;45(6):711-6. https://doi.org/10.1016/j.semarthrit.2015.11.002.Lenert A., Yao Q. Macrophage activation syndrome complicating adult onset Still's disease: A single center case series and comparison with literature. Semin Arthritis Rheum. 2016;45(6):711-6. https://doi.org/10.1016/j.semarthrit.2015.11.002.Wang C.R., Tsai Y.S., Ma I.C. Macrophage activation syndrome with lung involvement complicating adult-onset Still's disease. Neth J Med. 2018;76(10):442-4.Wang C.R., Tsai Y.S., Ma I.C. Macrophage activation syndrome with lung involvement complicating adult-onset Still's disease. Neth J Med. 2018;76(10):442-4.Peters A.T., Prickett M.H. Adult-onset Still's disease presenting as macrophage-activation syndrome with critical illness in the third trimester of pregnancy: A case report. Crit Care Explor. 2021;3(5):e0440. https://doi.org/10.1097/CCE.0000000000000440.Peters A.T., Prickett M.H. Adult-onset Still's disease presenting as macrophage-activation syndrome with critical illness in the third trimester of pregnancy: A case report. Crit Care Explor. 2021;3(5):e0440. https://doi.org/10.1097/CCE.0000000000000440.Hosoya S., Sadatsuki M., Izuka S. et al. A case of recurrence of adult-onset Still's disease in the third trimester: a case report and literature review. BMC Pregnancy Childbirth. 2021;21(1):163. https://doi.org/10.1186/s12884-021-03648-1.Hosoya S., Sadatsuki M., Izuka S. et al. A case of recurrence of adult-onset Still's disease in the third trimester: a case report and literature review. BMC Pregnancy Childbirth. 2021;21(1):163. https://doi.org/10.1186/s12884-021-03648-1.Артишевская Н.И., Раевнева Т.Г., Алексейчик С.Е. и др. Болезнь Стилла взрослых у беременной, осложненная синдромом активации макрофагов. Неотложная кардиология и кардиоваскулярные риски. 2020;4(1):877-81.Artishevskaia N.I., Rayeuneva T.G., Aliakseitshik S.E. et al. Adult-onset Still's disease in pregnancy complicated by macrophage activation syndrome. [Bolezn' Stilla vzroslyh u beremennoj, oslozhnennaya sindromom aktivacii makrofagov]. Neotlozhnaya kardiologiya i kardiovaskulyarnye riski. 2020;4(1):877-81. (In Russ.).Ravelli A., Minoia F., Davi S. et al.; Paediatric Rheumatology International Trials Organisation; Childhood Arthritis and Rheumatology Research Alliance; Pediatric Rheumatology Collaborative Study Group; Histiocyte Society. 2016 Classification criteria for macrophage activation syndrome complicating systemic juvenile idiopathic arthritis: A European League Against Rheumatism/American College of Rheumatology/Paediatric Rheumatology International Trials Organisation Collaborative Initiative. Arthritis Rheumatol. 2016;68(3):566-76. https://doi.org/10.1002/art.39332.Ravelli A., Minoia F., Davi S. et al.; Paediatric Rheumatology International Trials Organisation; Childhood Arthritis and Rheumatology Research Alliance; Pediatric Rheumatology Collaborative Study Group; Histiocyte Society. 2016 Classification criteria for macrophage activation syndrome complicating systemic juvenile idiopathic arthritis: A European League Against Rheumatism/American College of Rheumatology/Paediatric Rheumatology International Trials Organisation Collaborative Initiative. Arthritis Rheumatol. 2016;68(3):566-76. https://doi.org/10.1002/art.39332.Костик М.М., Дубко М.Ф., Масалова В.В. и др. Современные подходы к диагностике и лечению синдрома активации макрофагов у детей с ревматическими заболеваниями. Современная ревматология. 2015;(1):55-9. https://doi.org/10.14412/1996-7012-2015-1-55-59.Kostik M.M., Dubko M.F., Masalova V.V. et al. Current approaches to diagnosing and treating macrophage activation syndrome in children with rheumatic diseases. [Sovremennye podhody k diagnostike i lecheniyu sindroma aktivacii makrofagov u detej s revmaticheskimi zabolevaniyami]. Sovremennaya revmatologiya. 2015;(1):55-9. (In Russ.). https://doi.org/10.14412/1996-7012-2015-1-55-59.Pouchot J., Sampalis J.S., Beaudet F. et al. Adult Still's disease: Manifestations, disease course, and outcome in 62 patients. Medicine. 1991;70(2):118-36.Pouchot J., Sampalis J.S., Beaudet F. et al. Adult Still's disease: Manifestations, disease course, and outcome in 62 patients. Medicine. 1991;70(2):118-36.Rau M., Schiller M., Krienke S. et al. Clinical manifestations but not cytokine profiles differentiate adult-onset Still's disease and sepsis. J Rheumatol. 2010;37(11):2369-76. https://doi.org/10.14412/1996-7012-2015-1-55-5910.3899/jrheum.100247.Rau M., Schiller M., Krienke S. et al. Clinical manifestations but not cytokine profiles differentiate adult-onset Still's disease and sepsis. J Rheumatol. 2010;37(11):2369-76. https://doi.org/10.14412/1996-7012-2015-1-55-5910.3899/jrheum.100247.Wang R., Li T., Ye S. et al. Macrophage activation syndrome associated with adult-onset Still's disease: a multicenter retrospective analysis. Clin Rheumatol. 2020;39(8):2379-86. https://doi.org/10.14412/1996-7012-2015-1-55-5910.1007/s10067-020-04949-0.Wang R., Li T., Ye S. et al. Macrophage activation syndrome associated with adult-onset Still's disease: a multicenter retrospective analysis. Clin Rheumatol. 2020;39(8):2379-86. https://doi.org/10.14412/1996-7012-2015-1-55-5910.1007/s10067-020-04949-0.Клинические рекомендации - Юношеский артрит с системным началом - 2021-2022-2023 (29.06.2021). М.: Министерство здраво¬охранения Российской Федерации, 2021. 192 с. Режим доступа: https://cr.minzdrav.gov.ru/recomend/26_2. [Дата обращения: 01.02.2024].Clinical guidelines - Juvenile arthritis with systemic onset - 2021-2022-2023 (29.06.2021). [Klinicheskie rekomendacii - Yunosheskij artrit s sistemnym nachalom - 2021-2022-2023 (29.06.2021)]. Moscow: Ministerstvo zdravoohraneniya Rossijskoj Federacii, 2021. 192 p. Available at: https://cr.minzdrav.gov.ru/recomend/26_2. [Accessed: 01.02.2024].

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