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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">In recent years&#44; pulmonary diseases &#40;LD&#41; such as pulmonary arterial hypertension &#40;PAH&#41;&#44; interstitial lung disease &#40;ILD&#41; and pulmonary alveolar proteinosis &#40;PAP&#41; have been reported as life-threatening complications of systemic juvenile idiopathic arthritis &#40;sJIA&#41;&#46; In sJIA-LD&#44; a cytokine storm caused by interleukin &#40;IL&#41;-18 and interferon &#40;IFN&#41;-&#947; is considered to affect the differentiation of lung macrophages&#46; Potential new therapeutic options have included janus kinase inhibitors effective on IFN-&#947;-induced pathways&#46; We report a rare case of sJIA-LD and its treatment with januse kinase inhibitors&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">A 2-year-old male patient presented with fever for the last 15 days&#44; maculopapular rashes and right knee swelling&#46; Physical examination revealed right knee arthritis and splenomegaly&#46; Laboratory tests showed leukocytosis&#44; thrombocytosis&#44; anemia and elevated acute phase reactants&#46; Infections and malignancy were excluded&#46; Based on persistent fever&#44; maculopapular rash&#44; arthritis&#44; splenomegaly the patient was diagnosed with sJIA according to the International League of Associations for Rheumatology &#40;ILAR&#41; criteria&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">2</span></a> Steroid treatment was started&#46; Due to the recurrence of fever and elevated ESR and CRP in the patient&#39;s clinical progression despite having initially responded to steroid&#44; anakinra &#40;1&#8211;2<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;day&#41; was incorporated into his treatment&#46; Anakinra was switched to canakinumab &#40;4&#8211;8<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;month&#41; because of the difficulties experienced with daily injections&#46; In 1 year and 6 months the disease was clinically and laboratory activated three times&#46; His follow-ups were interrupted by his parents during the COVID-19 pandemic&#46; After 19 months&#44; he was admitted with complaints of fever&#44; rash&#44; cough&#44; swelling fingers&#46; The patient had maculopapular rashes in the extremities&#44; hepatosplenomegaly&#44; reduced respiratory sounds in the lower zones of the lungs&#44; widespread rales&#44; and clubbing &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; There was no need for respiratory support&#46; Oxygen saturation measurement by pulse oximetry was 98&#37;&#46; His posteroanterior thorax radiography revealed diffuse bilateral reticular opacity increase &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#44; and thoracic computed tomography &#40;CT&#41; showed diffuse interlobular septal thickening&#44; fibroelectatic retractions&#44; honeycomb pattern&#44; peribronchovascular thickening and pleural effusion &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; Bronchoalveolar lavage &#40;BAL&#41; infection screens were negative&#46; BAL cytology showed lipid-laden macrophages with PAS staining&#44; suggesting PAP&#46; Lung biopsy showed interstitial fibrosis in the subpleural area and cholesterol clefts in the alveoli&#46; Granuloma&#44; lymphoid aggregates&#44; follicular bronchiolitis-like morphology were not detected &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; The findings were diagnosed as sJIA-LD&#46; The limitations of our report include the fact that IFN signature&#44; IL-18 levels and lymphocyte subgroups in BAL cytology were could not be measured&#46; Pulse methylprednisolone was started and treatment was continued with 2<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;day prednisolone&#46; Canakinumab was discontinued and tofacitinib &#40;5<span class="elsevierStyleHsp" style=""></span>mg&#47;day&#41; was started&#46; At 1 month of tofacitinib treatment&#44; there were significant improvements in effort dyspnea and cough&#46; In the fifth month of tofacitinib treatment despite thoracic tomography showing no significant regression in pathologic findings except pleural effusion&#44; the patient was asymptomatic and respiratory sounds were normal&#46; The patient&#39;s follow-up continues with tofacitinib and steroids&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">In conclusion&#44; sJIA-LD is a life-threatening complication with unclear treatment options and a poor prognosis&#46; Our patient had multiple risk factors that may lead to sJIA-LD&#44; including early age&#44; recurrent macrophage activation syndrome attacks&#44; IL-1 inhibitor usage&#44; and uncontrolled systemic inflammation&#46;<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">3&#44;4</span></a> It is very important to keep in mind that LD may develop in patients with sJIA who have similar risk factors&#46; Potential new treatment options are janus kinase inhibitors acting on IFN-&#947;-induced pathways&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Informed Consent</span><p id="par0020" class="elsevierStylePara elsevierViewall">Written informed consent was obtained from the parents for publication of this case report and any accompanying images&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Funding</span><p id="par0025" class="elsevierStylePara elsevierViewall">An honorarium&#44; grant&#44; or other form of payment was not given to any author to produce the manuscript&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Authors&#8217; Contributions</span><p id="par0030" class="elsevierStylePara elsevierViewall">All authors made substantial contributions to the conception or design of the work&#44; have approved the final manuscript&#44; and takes full responsibility for the manuscript&#46; MMK&#44; EC reviewed and revised the manuscript&#44; MMK&#44; EC and GC contributed to the writing of the manuscript&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Conflict of Interest</span><p id="par0035" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span></span>"
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Journal Information
Vol. 60. Issue 6.
Pages 374-375 (June 2024)
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Vol. 60. Issue 6.
Pages 374-375 (June 2024)
Case Report
Lung Disease: A Rare but Devastating Complication of Systemic Juvenile Idiopathic Arthritis and the Use of Tofacitinib as a Treatment
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Melike Mehveş Kaplana,
Corresponding author
melikemehves@gmail.com

Corresponding author.
, Güzin Cinelb, Elif Çelikela
a Division of Pediatric Rheumatology, Department of Pediatrics, University of Health Sciences, Ankara City Hospital, Turkey
b Division of Pediatric Pulmonology, Department of Pediatrics, University of Health Sciences, Ankara City Hospital, Turkey
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