Two Sisters with Colchicine-Resistant form of Familial Mediterranean Fever on the Canakinumab Therapy: Clinical Case

Background. Colchicine-resistant cases of familial Mediterranean fever (FMF) are associated with high risk of kidney amyloidosis and severe episodes of the disease with pyretic fever, arthritis, pleurisies, pericarditis. It is difficult to achieve disease control in colchicine-resistant patients and...

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Main Authors: Elena A. Ligostaeva, Nadezhda A. Tsurikova, Irina A. Tsiganok, Nataliya N. Kobzeva, Vadim Yu. Avdeenko
Format: Article
Language:English
Published: "Paediatrician" Publishers LLC 2020-01-01
Series:Вопросы современной педиатрии
Subjects:
Online Access:https://vsp.spr-journal.ru/jour/article/view/2234
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author Elena A. Ligostaeva
Nadezhda A. Tsurikova
Irina A. Tsiganok
Nataliya N. Kobzeva
Vadim Yu. Avdeenko
author_facet Elena A. Ligostaeva
Nadezhda A. Tsurikova
Irina A. Tsiganok
Nataliya N. Kobzeva
Vadim Yu. Avdeenko
author_sort Elena A. Ligostaeva
collection DOAJ
description Background. Colchicine-resistant cases of familial Mediterranean fever (FMF) are associated with high risk of kidney amyloidosis and severe episodes of the disease with pyretic fever, arthritis, pleurisies, pericarditis. It is difficult to achieve disease control in colchicine-resistant patients and it is required to use genetically engineered biologic drugs.Clinical Case Description. The observation of the family with severe course of FFL is presented. The successful use of canakinumab (monoclonal antibody to interleukin-1) in sisters with colchicine-resistant form of disease is described. Fever, articular and abdominal syndromes were completely reversed after 4 weeks of treatment as well as normalisation of laboratory tests was noted. Laboratory indexes of disease activity (erythrocyte sedimentation rate and C-reactive protein level) have stayed in reference ranges after 32 weeks of therapy, no new episodes of the disease were recorded. There were no adverse effect on the canakinumab therapy during the observation period (32 weeks).Conclusion. The high canakinumab efficacy in patients with severe colchicine-resistant forms of FMF is shown.
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spelling doaj.art-a5ef08fc00d648debad1630c927267402023-09-03T09:26:50Zeng"Paediatrician" Publishers LLCВопросы современной педиатрии1682-55271682-55352020-01-0118536236810.15690/vsp.v18i5.20601838Two Sisters with Colchicine-Resistant form of Familial Mediterranean Fever on the Canakinumab Therapy: Clinical CaseElena A. Ligostaeva0Nadezhda A. Tsurikova1Irina A. Tsiganok2Nataliya N. Kobzeva3Vadim Yu. Avdeenko4Областная детская клиническая больницаОбластная детская клиническая больницаОбластная детская клиническая больницаОбластная детская клиническая больницаОбластная детская клиническая больницаBackground. Colchicine-resistant cases of familial Mediterranean fever (FMF) are associated with high risk of kidney amyloidosis and severe episodes of the disease with pyretic fever, arthritis, pleurisies, pericarditis. It is difficult to achieve disease control in colchicine-resistant patients and it is required to use genetically engineered biologic drugs.Clinical Case Description. The observation of the family with severe course of FFL is presented. The successful use of canakinumab (monoclonal antibody to interleukin-1) in sisters with colchicine-resistant form of disease is described. Fever, articular and abdominal syndromes were completely reversed after 4 weeks of treatment as well as normalisation of laboratory tests was noted. Laboratory indexes of disease activity (erythrocyte sedimentation rate and C-reactive protein level) have stayed in reference ranges after 32 weeks of therapy, no new episodes of the disease were recorded. There were no adverse effect on the canakinumab therapy during the observation period (32 weeks).Conclusion. The high canakinumab efficacy in patients with severe colchicine-resistant forms of FMF is shown.https://vsp.spr-journal.ru/jour/article/view/2234детисемейная средиземноморская лихорадкаклинический случайпериодическая болезньколхицинканакинумабинтерлейкин 1активность болезнинежелательные реакции
spellingShingle Elena A. Ligostaeva
Nadezhda A. Tsurikova
Irina A. Tsiganok
Nataliya N. Kobzeva
Vadim Yu. Avdeenko
Two Sisters with Colchicine-Resistant form of Familial Mediterranean Fever on the Canakinumab Therapy: Clinical Case
Вопросы современной педиатрии
дети
семейная средиземноморская лихорадка
клинический случай
периодическая болезнь
колхицин
канакинумаб
интерлейкин 1
активность болезни
нежелательные реакции
title Two Sisters with Colchicine-Resistant form of Familial Mediterranean Fever on the Canakinumab Therapy: Clinical Case
title_full Two Sisters with Colchicine-Resistant form of Familial Mediterranean Fever on the Canakinumab Therapy: Clinical Case
title_fullStr Two Sisters with Colchicine-Resistant form of Familial Mediterranean Fever on the Canakinumab Therapy: Clinical Case
title_full_unstemmed Two Sisters with Colchicine-Resistant form of Familial Mediterranean Fever on the Canakinumab Therapy: Clinical Case
title_short Two Sisters with Colchicine-Resistant form of Familial Mediterranean Fever on the Canakinumab Therapy: Clinical Case
title_sort two sisters with colchicine resistant form of familial mediterranean fever on the canakinumab therapy clinical case
topic дети
семейная средиземноморская лихорадка
клинический случай
периодическая болезнь
колхицин
канакинумаб
интерлейкин 1
активность болезни
нежелательные реакции
url https://vsp.spr-journal.ru/jour/article/view/2234
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AT irinaatsiganok twosisterswithcolchicineresistantformoffamilialmediterraneanfeveronthecanakinumabtherapyclinicalcase
AT nataliyankobzeva twosisterswithcolchicineresistantformoffamilialmediterraneanfeveronthecanakinumabtherapyclinicalcase
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