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The authors have described the world’s first case of necrotizing sarcoid granulomatosis (NSG) in a 22-year-old woman with the clinical presentations of acute abdomen, which are associated with abdominal lymph nodal infiltration and necrosis, obvious constitutional disturbances (fever, nocturnal sweats, and significant weight loss), high inflammatory activity (anemia, leukocytosis, high erythrocyte sedimentation rates and C-reactive protein levels), the gradual appearance of splenic and hepatic necrotic foci, and infiltration into the lung and lacrimal glands with the development of unilateral uveitis. The patient underwent five surgical interventions, several needle biopsies for recurrent abdominal syndrome, and long-term antibiotic treatment for presumed sepsis, which had caused drug-induced hepatitis. Bacteriological examination of blood, puncture samples, and removed abdominal cavity tissues, serological tests, and immunomorphogical study of biopsy samples and removed tissues yielded negative results for the presence of bacterial, fungal, and tuberculosis infections. NSG was diagnosed on the basis of the systemic nature of the lesion, the presence of granulomas with severe abdominal lymph nodal necrosis and necrotizing granulomatous/lymphocytic vasculitis in the mesentery and removed spleen, as well as the absence of granulomas in the spleen, appendix, and biopsy materials of the liver, colonic mucosa, and parotid gland. Fludarabine therapy was first used in world practice due to the inefficient treatment with high-dose glucocorticoids and cyclophosphamide and to a disease relapse when reducing their doses. The paper gives a detailed review of the literature on the clinical, laboratory, radiological, and morphological manifestations of the disease, which allow the differential diagnosis of NSG with different variants of granulomatous lesions. Based on the 5-year follow-up of the patient and on the analysis of clinical, laboratory, radiological, and morphological changes, the authors uphold the concept that the disease is an independent nosological entity: necrotizing angiitis with sarcoid reactions, rather than the entity of nodular or classic sarcoidosis.
The authors have described the world’s first case of necrotizing sarcoid granulomatosis (NSG) in a 22-year-old woman with the clinical presentations of acute abdomen, which are associated with abdominal lymph nodal infiltration and necrosis, obvious constitutional disturbances (fever, nocturnal sweats, and significant weight loss), high inflammatory activity (anemia, leukocytosis, high erythrocyte sedimentation rates and C-reactive protein levels), the gradual appearance of splenic and hepatic necrotic foci, and infiltration into the lung and lacrimal glands with the development of unilateral uveitis. The patient underwent five surgical interventions, several needle biopsies for recurrent abdominal syndrome, and long-term antibiotic treatment for presumed sepsis, which had caused drug-induced hepatitis. Bacteriological examination of blood, puncture samples, and removed abdominal cavity tissues, serological tests, and immunomorphogical study of biopsy samples and removed tissues yielded negative results for the presence of bacterial, fungal, and tuberculosis infections. NSG was diagnosed on the basis of the systemic nature of the lesion, the presence of granulomas with severe abdominal lymph nodal necrosis and necrotizing granulomatous/lymphocytic vasculitis in the mesentery and removed spleen, as well as the absence of granulomas in the spleen, appendix, and biopsy materials of the liver, colonic mucosa, and parotid gland. Fludarabine therapy was first used in world practice due to the inefficient treatment with high-dose glucocorticoids and cyclophosphamide and to a disease relapse when reducing their doses. The paper gives a detailed review of the literature on the clinical, laboratory, radiological, and morphological manifestations of the disease, which allow the differential diagnosis of NSG with different variants of granulomatous lesions. Based on the 5-year follow-up of the patient and on the analysis of clinical, laboratory, radiological, and morphological changes, the authors uphold the concept that the disease is an independent nosological entity: necrotizing angiitis with sarcoid reactions, rather than the entity of nodular or classic sarcoidosis.
Прогресс в ревматологии, прежде всего, связан с внедрением генно-инженерных биологических препаратов (ГИБП), которые демонстрируют преимущества перед стандартными методами лечения в отношении как эффективности, так и безопасности. При артериите Такаясу вопросы применения ГИБП не разработаны. Среди перспективных направлений лечения рефрактерного артериита Такаясу рассматривают ингибирование интерлейкина (ИЛ)-6. Представлен собственный опыт эффективного применения тоцилизумаба (гуманизированные моноклональные антитела к ИЛ-6 рецептору) у пациентки 27 лет с артериитом Такаясу, резистентным к лечению глюкокортикоидами и цитостатиками (циклофосфан, метотрексат), неполным эффектом альтернативной терапии флударабином, непереносимостью циклофосфана и осложнениями лечения глюкокортикоидами. В результате лечения тоцилизумабом (600 мг внутривенно каждые 4 недели в течение 24-х месяцев) достигнута стойкая ремиссия артериита Такаясу, подтвержденная результатами позитронной эмиссионной томографии, и минимизирована доза глюкокортикоидов. Применение ГИБП, действие которых направлено на ингибирование ИЛ-6, можно рассматривать как перспективную инновационную стратегию лечения артериита Такаясу, в первую очередь при неэффективности стандартной терапии или ее плохой переносимости. Ключевые слова: артериит Такаясу, генно-инженерные биологические препараты, интерлейкин-6, тоцилизумаб Для цитирования: Антипова ВН, Солодовникова ЛВ, Бекетова ТВ. Эффективность ингибитора интерлейкина-6 тоцилизумаба при артериите Такаясу с недостаточным ответом на предшествующее лечение глюкокортикоидами, метотрексатом, циклофосфаном, флударабином (Клиническое наблюдение).
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