vspВопросы современной педиатрииCurrent Pediatrics1682-55271682-5535Издательство «ПедиатрЪ»10.15690/vsp.v23i5.2807vsp-3612Research ArticleКЛИНИЧЕСКОЕ НАБЛЮДЕНИЕCLINICAL OBSERVATIONSЛечение тофацитинибом подростка с генерализованной формой витилиго: клинический случайManagement of Generalized Vitiligo in Adolescent with Tofacitinib: Clinical Casehttps://orcid.org/0000-0002-8232-8936АмбарчянЭ. Т.AmbarchyanEduard T.

Амбарчян Эдуард Тигранович, кандидат медицинских наук, заведующий отделением дерматологии НИИ педиатрии и охраны здоровья детей

117593, Москва, Литовский бульвар, д. 1а, тел.: +7 (915) 400-00-02

Moscow

edo_amb@mail.ruhttps://orcid.org/0000-0003-3003-9398КузьминоваА. Д.KuzminovaAnastasiya D.

Москва

Moscow

https://orcid.org/0000-0002-6760-3119ИванчиковВ. В.IvanchikovVladislav V.

Москва

Moscow

https://orcid.org/0000-0002-2063-6351РоговаА. А.RogovaAleksandra A.

Москва

Moscow

НИИ педиатрии и охраны здоровья детей НКЦ №2 ФГБНУ «РНЦХ им. акад. Б.В. Петровского»РоссияResearch Institute of Pediatrics and Children’s Health in Petrovsky National Research Centre of SurgeryRussian FederationРНИМУ им. Н.И. ПироговаРоссияPirogov Russian National Research Medical UniversityRussian Federation202431102024235376383Copyright © Амбарчян Э.Т., Кузьминова А.Д., Иванчиков В.В., Рогова А.А., 20242024Амбарчян Э.Т., Кузьминова А.Д., Иванчиков В.В., Рогова А.А.Ambarchyan E.T., Kuzminova A.D., Ivanchikov V.V., Rogova A.A.Данная работа распространяется под лицензией Creative Commons Attribution 4.0.This work is licensed under a Creative Commons Attribution 4.0 License.https://vsp.spr-journal.ru/jour/article/view/3612

Обоснование. Витилиго — депигментирующее заболевание кожи, характеризующееся избирательной потерей меланоцитов, что приводит к образованию типичных белых пятен. Существует большое количество теорий этиологии витилиго, среди которых генетическая, аутоиммунная, нейрогенная, аутовоспалительная, теория окислительного стресса и многие другие. Общепризнанная доминирующая роль отводится концепции аутоиммунной природы витилиго. В терапии витилиго традиционно применяются различные методы фототерапии, топические и системные глюкокортикоиды (ГК), ингибиторы кальциневрина. В последнее время свою эффективность в лечении витилиго демонстрируют ингибиторы Янус-киназ (Janus kinase; JAK). Описание клинического случая. У подростка мужского пола, возраста 13 лет, жалобы на участки гипопигментации на коже лица, туловища и конечностей, возникшие после активной солнечной инсоляции во время отдыха летом. На основании клинической картины дерматологом был установлен диагноз «витилиго». В лечении применялись топические ГК 3-го класса активности, а также курс локальной узкополосной средневолновой фототерапии 311 нм № 30 без значимого эффекта. Пациент поступил в отделение дерматологии НИИ педиатрии и охраны здоровья детей НКЦ №2 ФГБНУ «РНЦХ им. акад. Б.В. Петровского», где в связи с неэффективностью применяемого ранее лечения и генерализованной формой заболевания была инициирована терапия ингибитором JAK тофацитинибом. Заключение. Лечение витилиго ингибиторами JAK, в частности тофацитинибом, является перспективной методикой и может привести к достижению значимого клинического эффекта с профилем безопасности, сопоставимым с традиционными методами терапии данной патологии.

Background. Vitiligo is a depigmenting skin disease characterized by selective loss of melanocytes leading to development of typical white spots. There are various theories on vitiligo etiology: genetic, autoimmune, neurogenic, autoinflammatory, oxidative stress theory, and many others. Generally accepted dominant role is given to the concept of its autoimmune nature. Vitiligo management traditionally includes different methods of phototherapy, topical and systemic glucocorticoids (GC), and calcineurin inhibitors. Recently, Janus kinase inhibitors (JAK) have shown its efficacy in treatment of vitiligo. Clinical case description. 13-year-old male adolescent has complaints of hypopigmentation areas on the skin of face, trunk and limbs that appeared after active solar insolation during summer vacation. Dermatologist has determined a diagnosis of vitiligo according to clinical picture. Topical GC of the 3rd activity class were used for treatment, as well as course of local narrow-band medium-wave photodynamic therapy (311 nm, No. 30) with no significant effect. The patient was admitted to the dermatology department of Research Institute of Pediatrics and Children’s Health in Petrovsky National Research Centre of Surgery where therapy with JAK inhibitor, tofacitinib, was initiated due to inefficacy of previous treatment and the generalized form of the disease. Conclusion. Management of vitiligo with JAK inhibitors, in particular tofacitinib, is a promising method and it can lead to significant clinical effect with safety profile comparable to conventional therapies for this pathology.

витилигоингибиторы JAKтофацитинибдетиподросткиvitiligoJAK inhibitorstofacitinibchildrenadolescentsОтсутствует.Not declared.ReferencesMurashkin NN, Savelova AA, Ivanov RA, et al. Etiopathogenetic Similarities of Combined Forms of Localized Scleroderma and Vitiligo. Voprosy sovremennoi pediatrii — Current Pediatrics. 2020;19(6):452–459. (In Russ). doi: https://doi.org/10.15690/vsp.v19i6.2147Murashkin NN, Savelova AA, Ivanov RA, et al. Etiopathogenetic Similarities of Combined Forms of Localized Scleroderma and Vitiligo. Voprosy sovremennoi pediatrii — Current Pediatrics. 2020;19(6):452–459. (In Russ). doi: https://doi.org/10.15690/vsp.v19i6.2147Chellapandian N, Boopathi D. Oral vitiligo: a predominant spread from oral mucosa. BMJ Case Rep. 2023;16(10):e257609. doi: https://doi.org/10.1136/bcr-2023-257609Chellapandian N, Boopathi D. Oral vitiligo: a predominant spread from oral mucosa. BMJ Case Rep. 2023;16(10):e257609. doi: https://doi.org/10.1136/bcr-2023-257609Ezzedine K, Eleftheriadou V, Whitton M, van Geel N. Vitiligo. Lancent. 2015:386(9988):74–84. doi: https://doi.org/10.1016/s0140-6736(14)60763-7Ezzedine K, Eleftheriadou V, Whitton M, van Geel N. Vitiligo. Lancent. 2015:386(9988):74–84. doi: https://doi.org/10.1016/s0140-6736(14)60763-7Sitek JC. Vitiligo — loss of cutaneous pigmentation. Tidsskr Nor Laegeforen. 2006;126(18):2370–2372.Sitek JC. Vitiligo — loss of cutaneous pigmentation. Tidsskr Nor Laegeforen. 2006;126(18):2370–2372.Rashighi M, Harris JE. Vitiligo Pathogenesis and Emerging Treatments. Dermatol Clin. 2017;35(2):257–265. doi: https://doi.org/10.1016/j.det.2016.11.014Rashighi M, Harris JE. Vitiligo Pathogenesis and Emerging Treatments. Dermatol Clin. 2017;35(2):257–265. doi: https://doi.org/10.1016/j.det.2016.11.014Komnitski M, Komnitski A, Komnitski Junior A, Silva de Castro CC. Partial repigmentation of vitiligo with tofacitinib, without exposure to ultraviolet radiation. An Bras Dermatol. 2020;Komnitski M, Komnitski A, Komnitski Junior A, Silva de Castro CC. Partial repigmentation of vitiligo with tofacitinib, without exposure to ultraviolet radiation. An Bras Dermatol. 2020;Damsky W, King BA. JAK inhibitors in dermatology: The promise of a new drug class. J Am Acad Dermatol. 2017;76(4):736–744. doi: https://doi.org/10.1016/j.jaad.2016.12.005Damsky W, King BA. JAK inhibitors in dermatology: The promise of a new drug class. J Am Acad Dermatol. 2017;76(4):736–744. doi: https://doi.org/10.1016/j.jaad.2016.12.005Kussainova A, Kassym L, Akhmetova A, et al. Vitiligo and anxiety: A systematic review and meta-analysis. PLoS One. 2020;15(11): e0241445. doi: https://doi.org/10.1371/journal.pone.0241445Kussainova A, Kassym L, Akhmetova A, et al. Vitiligo and anxiety: A systematic review and meta-analysis. PLoS One. 2020;15(11): e0241445. doi: https://doi.org/10.1371/journal.pone.0241445Behrangi E, Barough MS, Khoramdad M, et al. Efficacy and safety of tofacitinib for treatment of alopecia areata in children: A systematic review and meta-analysis. J Cosmet Dermatol. 2022;21(12): 6644–6652. doi: https://doi.org/10.1111/jocd.15425Behrangi E, Barough MS, Khoramdad M, et al. Efficacy and safety of tofacitinib for treatment of alopecia areata in children: A systematic review and meta-analysis. J Cosmet Dermatol. 2022;21(12): 6644–6652. doi: https://doi.org/10.1111/jocd.15425Huang J, Li T, Tan Z, et al. Effectiveness of Tofacitinib in Pre-adolescent Alopecia Areata: A Retrospective Case Series and Literature Review. Acta Derm Venereol. 2023;103:adv13418. doi: https://doi.org/10.2340/actadv.v103.13418Huang J, Li T, Tan Z, et al. Effectiveness of Tofacitinib in Pre-adolescent Alopecia Areata: A Retrospective Case Series and Literature Review. Acta Derm Venereol. 2023;103:adv13418. doi: https://doi.org/10.2340/actadv.v103.13418AlMutairi N, Nour T. Tofacitinib in Pediatric Psoriasis: An Open-Label Trial to Study Its Safety and Efficacy in Children. Dermatology. 2020;236(3):191–198. doi: https://doi.org/10.1159/000503062AlMutairi N, Nour T. Tofacitinib in Pediatric Psoriasis: An Open-Label Trial to Study Its Safety and Efficacy in Children. Dermatology. 2020;236(3):191–198. doi: https://doi.org/10.1159/000503062Berbert Ferreira S, Berbert Ferreira R, Neves Neto AC, et al. Topical Tofacitinib: A Janus Kinase Inhibitor for the Treatment of Vitiligo in an Adolescent Patient. Case Rep Dermatol. 2021;13(1):190–194. doi: https://doi.org/10.1159/000513938Berbert Ferreira S, Berbert Ferreira R, Neves Neto AC, et al. Topical Tofacitinib: A Janus Kinase Inhibitor for the Treatment of Vitiligo in an Adolescent Patient. Case Rep Dermatol. 2021;13(1):190–194. doi: https://doi.org/10.1159/000513938Sheikh A, Rafique W, Owais R, et al. FDA approves Ruxolitinib (Opzelura) for Vitiligo Therapy: A breakthrough in the field of dermatology. Ann Med Surg (Lond). 2022;81:104499. doi: https://doi.org/10.1016/j.amsu.2022.104499Sheikh A, Rafique W, Owais R, et al. FDA approves Ruxolitinib (Opzelura) for Vitiligo Therapy: A breakthrough in the field of dermatology. Ann Med Surg (Lond). 2022;81:104499. doi: https://doi.org/10.1016/j.amsu.2022.104499Marchioro HZ, Silva de Castro CC, Fava VM, et al. Update on the pathogenesis of vitiligo. An Bras Dermatol. 2022;97(4):478-490. doi: https://doi.org/10.1016/j.abd.2021.09.008Marchioro HZ, Silva de Castro CC, Fava VM, et al. Update on the pathogenesis of vitiligo. An Bras Dermatol. 2022;97(4):478-490. doi: https://doi.org/10.1016/j.abd.2021.09.008Ezzedine K, Lim HW, Suzuki T, et al. Revised classification/ nomenclature of vitiligo and related issues: the Vitiligo Global Issues Consensus Conference. Pigment Cell Melanoma Res. 2012;25(3):E1–E13. doi: https://doi.org/10.1111/j.1755-148X.2012.00997.xEzzedine K, Lim HW, Suzuki T, et al. Revised classification/ nomenclature of vitiligo and related issues: the Vitiligo Global Issues Consensus Conference. Pigment Cell Melanoma Res. 2012;25(3):E1–E13. doi: https://doi.org/10.1111/j.1755-148X.2012.00997.xEzzedine K, Soliman AM, Li C, et al. Comorbidity Burden Among Patients with Vitiligo in the United States: A Large-Scale Retrospective Claims Database Analysis. Dermatol Ther (Heidelb). 2023;13(10): 2265–2277. doi: https://doi.org/10.1007/s13555-023-01001-2Ezzedine K, Soliman AM, Li C, et al. Comorbidity Burden Among Patients with Vitiligo in the United States: A Large-Scale Retrospective Claims Database Analysis. Dermatol Ther (Heidelb). 2023;13(10): 2265–2277. doi: https://doi.org/10.1007/s13555-023-01001-2Eleftheriadou V, Atkar R, Batchelor J, et al. British Association of Dermatologists guidelines for the management of people with vitiligo 2021. Br J Dermatol. 2022;186(1):18–29. doi: https://doi.org/10.1111/bjd.20596Eleftheriadou V, Atkar R, Batchelor J, et al. British Association of Dermatologists guidelines for the management of people with vitiligo 2021. Br J Dermatol. 2022;186(1):18–29. doi: https://doi.org/10.1111/bjd.20596Lasheen RM, Elsheikh MN, Fawzy MM, Tomoum MO. Audiovestibular Abnormalities in Patients With Vitiligo: A Prospective Case-control Study. Otol Neurotol. 20211;42(5):e514–e520. doi: https://doi.org/10.1097/MAO.0000000000003044Lasheen RM, Elsheikh MN, Fawzy MM, Tomoum MO. Audiovestibular Abnormalities in Patients With Vitiligo: A Prospective Case-control Study. Otol Neurotol. 20211;42(5):e514–e520. doi: https://doi.org/10.1097/MAO.0000000000003044Genedy R, Assal S, Gomaa A, et al. Ocular and auditory abnormalities in patients with vitiligo: a case-control study. Clin Exp Dermatol. 2021;46(6):1058–1066. doi: https://doi.org/10.1111/ced.14649Genedy R, Assal S, Gomaa A, et al. Ocular and auditory abnormalities in patients with vitiligo: a case-control study. Clin Exp Dermatol. 2021;46(6):1058–1066. doi: https://doi.org/10.1111/ced.14649Bergqvist C, Ezzedine K. Vitiligo: A focus on pathogenesis and its therapeutic implications. J Dermatol. 2021;48(3):252–270. doi: https://doi.org/10.1111/1346-8138.15743Bergqvist C, Ezzedine K. Vitiligo: A focus on pathogenesis and its therapeutic implications. J Dermatol. 2021;48(3):252–270. doi: https://doi.org/10.1111/1346-8138.15743Craiglow BG, King BA. Tofacitinib Citrate for the Treatment of Vitiligo: A Pathogenesis-Directed Therapy. JAMA Dermatol. 2015;151(10):1110–1112. doi: https://doi.org/10.1001/jamadermatol.2015.1520Craiglow BG, King BA. Tofacitinib Citrate for the Treatment of Vitiligo: A Pathogenesis-Directed Therapy. JAMA Dermatol. 2015;151(10):1110–1112. doi: https://doi.org/10.1001/jamadermatol.2015.1520Kubelis-López DE, Zapata-Salazar NA, Said-Fernández SL, et al. Updates and new medical treatments for vitiligo (Review). Exp Ther Med. 2021;22(2):797. doi: https://doi.org/10.3892/etm.2021.10229Kubelis-López DE, Zapata-Salazar NA, Said-Fernández SL, et al. Updates and new medical treatments for vitiligo (Review). Exp Ther Med. 2021;22(2):797. doi: https://doi.org/10.3892/etm.2021.10229Miot HA, Criado PR, de Castro CCS, et al. JAK-STAT pathway inhibitors in dermatology. An Bras Dermatol. 2023;98(5):656–677. doi: https://doi.org/10.1016/j.abd.2023.03.001Miot HA, Criado PR, de Castro CCS, et al. JAK-STAT pathway inhibitors in dermatology. An Bras Dermatol. 2023;98(5):656–677. doi: https://doi.org/10.1016/j.abd.2023.03.001Sandborn WJ, Peyrin-Biroulet L, Quirk D, et al. Efficacy and Safety of Extended Induction With Tofacitinib for the Treatment of Ulcerative Colitis. Clin Gastroenterol Hepatol. 2022;20(8):1821–1830.e3. doi: https://doi.org/10.1016/j.cgh.2020.10.038Sandborn WJ, Peyrin-Biroulet L, Quirk D, et al. Efficacy and Safety of Extended Induction With Tofacitinib for the Treatment of Ulcerative Colitis. Clin Gastroenterol Hepatol. 2022;20(8):1821–1830.e3. doi: https://doi.org/10.1016/j.cgh.2020.10.038Khosrow-Khavar F, Kim SC, Lee H, et al. Tofacitinib and risk of cardiovascular outcomes: results from the Safety of TofAcitinib in Routine care patients with Rheumatoid Arthritis (STAR-RA) study. Ann Rheum Dis. 2022;81(6):798–804. doi: https://doi.org/10.1136/annrheumdis-2021-221915Khosrow-Khavar F, Kim SC, Lee H, et al. Tofacitinib and risk of cardiovascular outcomes: results from the Safety of TofAcitinib in Routine care patients with Rheumatoid Arthritis (STAR-RA) study. Ann Rheum Dis. 2022;81(6):798–804. doi: https://doi.org/10.1136/ annrheumdis-2021-221915Lima LM, Aurilio RB, Fonseca AR, et al. Tuberculosis in children and adolescents with rheumatic diseases using biologic agents: an integrative review. Rev Paul Pediatr. 2023;42:e2022084. doi: https://doi.org/10.1590/1984-0462/2024/42/2022084Lima LM, Aurilio RB, Fonseca AR, et al. Tuberculosis in children and adolescents with rheumatic diseases using biologic agents: an integrative review. Rev Paul Pediatr. 2023;42:e2022084. doi: https://doi.org/10.1590/1984-0462/2024/42/2022084Solimani F, Meier K, Ghoreschi K. Emerging Topical and Systemic JAK Inhibitors in Dermatology. Front Immunol. 2019;10:2847. doi: https://doi.org/10.3389/fimmu.2019.02847Solimani F, Meier K, Ghoreschi K. Emerging Topical and Systemic JAK Inhibitors in Dermatology. Front Immunol. 2019;10:2847. doi: https://doi.org/10.3389/fimmu.2019.02847

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