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    "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Takayasu&#39;s arteritis &#40;TA&#41;&#44; is a chronic idiopathic granulomatous inflammation of the aorta and its major branches&#44; which leads to wall thickening&#44; fibrosis&#44; stenosis&#44; occlusion and often aneurysm formation&#44; affecting mainly Asian women of child-bearing age&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">Crohn&#39;s disease &#40;CD&#41; is a chronic granulomatous inflammatory disease characterized by transmural inflammation and can involve any part of the gastrointestinal tract&#44; resulting in significant complications such as abscesses&#44; fistulas&#44; and strictures&#46; CD is often associated with extraintestinal complications&#44; with manifestations in various organs such as the skin&#44; eyes&#44; joints&#44; and cardiovascular system&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">Several case reports have noted TA and CD in the same patient&#44; though such coexistence is rare&#44; there could be a common pathophysiological link between these two diseases to induce chronic inflammation&#46; Herein&#44; we report a rare case of secondary hypertension&#44; a complex case of TA associated with CD with bad response to conventional immunosuppressive and biologic treatment&#44; after total proctocolectomy with end ileostomy&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Case report</span><p id="par0020" class="elsevierStylePara elsevierViewall">We report the case of a 25-year-old Caucasian female with CD and family history of her father having Ulcerative Colitis&#46; She was first diagnosed of inflammatory bowel disease &#40;IBD&#41; at the age of 8&#44; based on bloody stool&#44; ferropenic anemia and endoscopic findings&#44; which revealed macroscopic involvement from rectum to sigma&#44; with redness and multiple small erosions&#46; The biopsy findings were compatible with the diagnosis of severe active ulcerative colitis&#46; She remained with a good clinical response on oral and topical salicylates&#44; and only one severe flare-up episode required hospitalization and systemic corticosteroid treatment&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">In 2006&#44; at the age of 19&#44; the disease activity persisted high&#44; with anemia and inflammatory markers elevated&#44; erythrocyte sedimentation rate &#40;ESR&#41; 45<span class="elsevierStyleHsp" style=""></span>mm&#47;h &#91;range&#58; 3&#8211;20&#93; and C-reactive protein &#40;CRP&#41; 8&#46;2<span class="elsevierStyleHsp" style=""></span>mg&#47;dL &#91;range&#58; 0&#8211;0&#46;8&#93;&#46; The colonoscopy showed edema of the mucosa&#44; erythematous appearance&#44; petechiae&#44; superficial ulcerations and loss of haustrae&#44; moreover cytomegalovirus was undetectable&#46; She started treatment with azathioprine &#40;2&#46;5<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;day&#41; and oral prednisone &#40;60<span class="elsevierStyleHsp" style=""></span>mg&#47;day&#41;&#44; but she developed gastrointestinal intolerance to azathioprine&#46; We tried treatment with 6-mercaptopurine &#40;25<span class="elsevierStyleHsp" style=""></span>mg&#47;every 48<span class="elsevierStyleHsp" style=""></span>h&#41; but she also developed gastrointestinal intolerance&#46; We considered trying administration of anti-TNF-&#945; treatment for continuous inflammatory disease and the patient received infliximab &#40;IFX&#41; infusions at weeks 0&#44; 2 and 6 according to the conventional procedure &#40;5<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#41;&#44; and following&#44; every 8 weeks&#46; Although a partial clinical response was obtained&#44; biological &#40;CRP 4&#46;96<span class="elsevierStyleHsp" style=""></span>mg&#47;dL&#44; ESR 78<span class="elsevierStyleHsp" style=""></span>mm&#47;h&#44; fibrinogen 558<span class="elsevierStyleHsp" style=""></span>mg&#47;dL &#91;range&#58; 170&#8211;437&#93;&#41; and endoscopic activity persisted despite IFX treatment&#46; One year after she required corticosteroids again and became steroid-dependent with lack of response to IFX&#46; At that point&#44; we suggested her go on surgery&#44; though the patient refused&#46; The patient started a program of leucocytapheresis &#40;10 sessions in 2 months&#41;&#44; IFX infusions every 4 weeks and also oral and topical salicylates in high doses&#46; Six months later&#44; there was no improvement in the biological markers&#46; Thus&#44; anti-TNF therapy was changed from IFX to adalimumab &#40;40<span class="elsevierStyleHsp" style=""></span>mg&#47;week&#41;&#46; However&#44; acute phase reactants relapsed &#40;CRP 10&#46;7<span class="elsevierStyleHsp" style=""></span>mg&#47;dL&#44; ESR 81<span class="elsevierStyleHsp" style=""></span>mm&#47;h&#44; 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12&#8211;16&#93;&#44; hematocrit 35&#46;3&#37; &#91;range&#58; 37&#8211;47&#93;&#44; platelet count 302&#44;000&#47;&#956;L &#91;range&#58; 150&#44;000&#8211;400&#44;000&#93; and total protein 8&#46;8<span class="elsevierStyleHsp" style=""></span>g&#47;dL &#91;range&#58; 6&#46;6&#8211;8&#46;7&#93;&#46; Liver enzymes indices and coagulation were normal&#46; Renal test function showed an elevation of creatinin 1&#46;14<span class="elsevierStyleHsp" style=""></span>mg&#47;dL &#91;range&#58; 0&#46;1&#8211;0&#46;95&#93;&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">Ultrasonography revealed homogeneous wall thickening of right common carotid&#44; subclavian and axillar artery&#44; with a suspected significant stenosis of both renal arteries&#46; All consistent with long-standing hypertension of probable renal origin&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">Ophthalmologic examination did not reveal any signs of retinopathy&#46; Echocardiography only showed a moderated tricuspid incompetence&#44; which could estimate a pulmonary pressure of 35<span class="elsevierStyleHsp" style=""></span>mmHg&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">Subsequent computed tomography angiography scanning &#40;CTA&#41; revealed thickening of the wall at the juxtarenal aorta&#44; with extension into the proximal renal arteries and the celiac trunk&#44; in addition&#44; the right subclavian artery was markedly narrowed&#46; CTA findings were confirmed by magnetic resonance &#40;MR&#41; &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#44; B&#41;&#46; There was no evidence of stenosis in pulmonary arteries or aneurysms in the aorta&#46; Together&#44; the diagnosis of type IV TA was established &#40;according to Numano Classification&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0055" class="elsevierStylePara elsevierViewall">Due to the severe activity of the TA&#44; treatment with prednisone &#40;60<span class="elsevierStyleHsp" style=""></span>mg&#47;day&#41; and methotrexate &#40;10<span class="elsevierStyleHsp" style=""></span>mg&#47;week&#44; SC injection&#41; were started&#46; Decreases in CRP and ESR were achieved&#44; thus&#44; maintenance therapy was continued&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">Currently&#44; at the time of writing&#44; she does not have any active disease on prednisone 20<span class="elsevierStyleHsp" style=""></span>mg&#47;day&#44; which is tapering&#44; and methotrexate &#40;10<span class="elsevierStyleHsp" style=""></span>mg&#47;week&#41;&#46; Six months later&#44; her inflammatory markers remain low&#44; ESR 10<span class="elsevierStyleHsp" style=""></span>mm&#47;h&#44; CRP 0&#46;1<span class="elsevierStyleHsp" style=""></span>mg&#47;dL&#44; meanwhile&#44; due to her medically refractory renovascular hypertension&#44; percutaneous transluminal renal angioplasty &#40;PTRA&#41; was scheduled and performed 2 months later &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>A&#44; B&#41;&#44; with significant improvement in renal function and hypertension control one month after the procedure&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Discussion</span><p id="par0065" class="elsevierStylePara elsevierViewall">The case here presented shows the association of a difficult to treat CD and a TA&#46; The patient was first diagnosed with IBD in 1995&#44; followed by a diagnosis of ulcerative colitis 6 years later&#44; although finally confirmed to be affected with CD based on histologic findings 1 year ago &#40;A1 L2 B1 Montreal Classification&#41;&#44; a change of diagnosis that has been reported in some studies&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#8211;3</span></a> She was diagnosed of a TA recently&#44; despite being treated with different anti-TNF biological therapies in the past&#46; This unexpected coexistence could have influenced to the responsiveness to medical therapy and the atypical presentation of CD in this patient&#44; which have been reported in the literature&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">TA is known to be a type of idiopathic&#44; inflammatory&#44; granulomatous vasculitis mainly involving the aorta and its main branches&#46; The prevalence of this rare disease is higher in young Asian woman&#44;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> with 150 new cases occurring each year in Japan&#44; in comparison&#44; in Western countries its incidence has risen&#44; having 1&#8211;3 new cases per year per million population in United States and Europe&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> The 15-year survival rate is more than 80&#37;&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> Early diagnosis and treatment are very important for patients with TA to prevent vascular complications&#46;</p><p id="par0075" class="elsevierStylePara elsevierViewall">The chance for the two diseases to occur in the same patient is estimated to be 1 per 10 billion persons&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> The first case of CD associated with TA was described by Gateau et al&#46; in 1970&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> Since then&#44; many cases of this unusual association have been reported sporadically in the literature&#44; Kusunoki et al&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> reviewed and listed 37 cases of CD and TA reported in the literature&#44; founding that the age of onset of TA symptoms was simultaneous or later than that of CD in most of them&#46; The long term prognosis of each disease in patients with both is unknown&#46; Reny et al&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">11</span></a> reported that CD was present in 9&#37; of a group of 44 TA patients&#44; and pointed out that in these cases tended to be younger the age at diagnosis&#44; and also tended to have systemic symptoms more frequently than those with TA alone &#40;features such as Raynaud phenomenon&#44; weight loss&#44; fever&#44; arthralgias&#44; myalgias&#41;&#46; Moreover&#44; they noted that the distribution of vascular lesions in patients with both diseases were more frequent with the involvement of the supra aortic archs&#44; instead of aorta&#44; supraaortic trunk&#44; pulmonary arteries&#44; femoral&#44; renal&#44; mesenteric&#44; coronary&#44; vertebral arteries&#44; more common of patients with TA alone&#46;</p><p id="par0080" class="elsevierStylePara elsevierViewall">The pathogenesis of both diseases share a common feature of autoimmune origin&#44; highly suggestive of cell-mediated autoimmunity&#44; where includes predominantly CD4 lymphocytes&#44; dendritic cells and common inflammation mediators &#40;Interferon-&#947;&#44; TNF-&#945;&#44; cytokines&#44; oxidative and growth factors&#41;&#44; resulting in a granulomatous inflammation&#44;<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">12&#44;13</span></a> highlighted in the histologic findings of the present case&#46; The immunologic mechanism proposed is that TA and CD are primarily Th1 dominant conditions&#44; and the cytokines involved in this response may play an important role in the pathogenesis of both diseases<a class="elsevierStyleCrossRefs" href="#bib0070"><span class="elsevierStyleSup">14&#44;15</span></a>&#44; in this regard&#44; some studies highlighted the use of anti-TNF alpha&#44; as infliximab&#44; or more recently&#44; anti-interleukin-6 receptor &#40;IL-6R&#41; antibody&#44; tocilizumab&#44;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">16</span></a> might show beneficial therapeutic effects in both diseases&#44;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">17&#44;18</span></a> which had been effectively used&#44; even in a significant number of steroid-refractory TA&#46;<a class="elsevierStyleCrossRefs" href="#bib0080"><span class="elsevierStyleSup">16&#44;19&#44;20</span></a>&#46;</p><p id="par0085" class="elsevierStylePara elsevierViewall">Some reports have demonstrated the development of vasculitis as an adverse effect of infliximab&#44;<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">21</span></a> or the detection and extent of the vasculitis according to the degree of inflammation in IBD&#44;<a class="elsevierStyleCrossRef" href="#bib0110"><span class="elsevierStyleSup">22</span></a> although no relation with TA has been found&#44; it is worth consideration&#46;</p><p id="par0090" class="elsevierStylePara elsevierViewall">Various HLA haplotypes &#40;HLA-A24&#44; B52&#44; Dw12 and DR2&#41; are seen in patients with both Crohn&#39;s disease and TA&#46; A genetic link has not been identified yet&#46;<a class="elsevierStyleCrossRef" href="#bib0115"><span class="elsevierStyleSup">23</span></a></p><p id="par0095" class="elsevierStylePara elsevierViewall">Another possible cause is the mycobacterial infection&#44; serological evidence supports that there is an increase production of anti-galactosyl IgG antibody triggered in response to protein p38&#44; specific from mycobacteria in these patients&#46;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">17&#44;18&#44;24</span></a></p><p id="par0100" class="elsevierStylePara elsevierViewall">Despite these observations&#44; we still know little about features of the pathogenesis and etiology of these two diseases&#46;</p><p id="par0105" class="elsevierStylePara elsevierViewall">Attention should be paid to extraintestinal complications such as TA&#44; if there is a discrepancy between acute phase reactants and gastrointestinal symptoms in IBD patients&#46; For that reason&#44; a recent study suggested that non-invasive modalities including ultrasonography&#44; CT&#44; MRI&#44; and fluoro-<span class="elsevierStyleSmallCaps">d</span>-glucose-positron emission tomography &#40;FDG-PET&#41; can provide a diagnosis of TA early in the course of the disease&#46;<a class="elsevierStyleCrossRef" href="#bib0125"><span class="elsevierStyleSup">25</span></a></p><p id="par0110" class="elsevierStylePara elsevierViewall">Corticosteroids and immunosuppressive treatment could be used in both diseases&#44; although some patients may also require surgical or endovascular revascularization procedures&#46;</p><p id="par0115" class="elsevierStylePara elsevierViewall">In our case&#44; we used a combination therapy of oral corticosteroid at an initial dosage of 1<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;day and methotrexate &#40;10<span class="elsevierStyleHsp" style=""></span>mg&#47;m<span class="elsevierStyleSup">2</span>&#47;week&#41;&#46; After the initial 4 weeks&#44; we slowly tapered the steroid to 0&#46;5<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;day&#44; which was successful&#46;</p><p id="par0120" class="elsevierStylePara elsevierViewall">There are a few reports of revascularizations procedures&#44; being catheter-based interventional treatment the most used&#44; with encouraging results&#44;<a class="elsevierStyleCrossRef" href="#bib0130"><span class="elsevierStyleSup">26</span></a> which is preferably performed when the disease is not in an active phase&#46;</p><p id="par0125" class="elsevierStylePara elsevierViewall">In conclusion&#44; our report indicates that patient suffering from IBD&#44; can suffer from the rare coincidence for TA&#44; even more if there is a previous history of refractory CD and there is a discrepancy between inflammatory markers and gastrointestinal symptoms&#46; History taking&#44; physical examinations&#44; including measuring of bilateral blood pressure and pulses&#44; ischemic signs and detection of systolic ejection murmur&#44; are important for early diagnosis of TA in EC patients&#46; Therefore begin as soon as possible with an optimal choice of therapy&#44; induce and sustain remission&#44; reaching optimal circumstances for interventional revascularization procedures&#44; thus preventing severe complications&#44; such as renal hypertension in this case&#44; and raising awareness of a significant problem in the clinical setting&#46;</p></span></span>"
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          "en" => "<p id="spar0045" class="elsevierStyleSimplePara elsevierViewall">A&#46; Axial image from T1 contrast-enhanced MRI revealing a severe wall thickening of abdominal aorta with luminal narrowing &#40;arrow&#41;&#46; B&#46; Coronal reconstructed image from a contrast-enhanced MRI demonstrating serious stenosis of the renal arteries &#40;arrows&#41; and wall thickening of the juxtarenal abdominal aorta&#46;</p>"
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Clinical communication
Takayasu's arteritis associated with Crohn's disease
Y. Tung Chena,
Autor para correspondencia
yale.tung@comv.es

Corresponding author.
, J.A. Todoli Parraa, M. Iborra Colominob, J.L. Moll Guillenc, L.J. Martin Abadc, R. Garcia Marcosd, P. Nos Mateub
a Department of Internal Medicine, La Fe University and Polytechnic Hospital of Valencia, Valencia, Spain
b Department of Gastroenterology, La Fe University and Polytechnic Hospital of Valencia, Valencia, Spain
c Department of Nephrology, La Fe University and Polytechnic Hospital of Valencia, Valencia, Spain
d Department of Medical Imaging, Unit of Interventional Radiology, La Fe University and Polytechnic Hospital of Valencia, Valencia, Spain

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