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inflammatory chronic diseases &#40;such as sprondyloarthropathy&#44; inflammatory bowel disease and rheumatoid arthritis&#41;&#44; periodic fever syndromes&#44; among other examples&#46;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">1&#8211;4</span></a> Clinical and laboratory manifestations depend on the type of amyloidosis and may include&#44; for example&#44; easy bruising&#44; macroglossia&#44; signs and symptoms of heart failure or arrhythmias&#44; hepatomegaly&#44; coagulation disorders or nephrotic proteinuria&#46;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">1&#8211;3</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We present a case of an older woman with nephrotic syndrome and a previously non clarified inflammatory peripheral arthropathy&#44; with the latter becoming responsible for AA amyloidosis&#46; This 48-month follow-up proves the need and effectiveness of properly treating the underlying disease&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">O&#46; G&#46; D&#46;&#44; a 78-year-old caucasian woman with a personal history of seronegative peripheral polyarthritis&#44; poor therapeutic compliance and corticoid-induced diabetes mellitus&#44; was admitted for etiological study of constitutional syndrome&#44; inflammatory arthralgias&#44; diarrhea and anasarca refractory to oral diuretics&#46; Usual medication included methotrexate&#44; deflazacorte&#44; esomeprazole and tapentadol&#44; folic acid&#44; calcium carbonate plus cholecalciferol and insulin&#46; Denied drug allergies&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">The complementary study revealed anemia &#40;normocytic&#44; normochromic&#41; of 9&#46;2<span class="elsevierStyleHsp" style=""></span>g&#47;dL&#44; normal leukogram&#44; thrombocytosis &#40;720<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>10&#94;9&#47;L&#41;&#44; pCr of 1&#46;3<span class="elsevierStyleHsp" style=""></span>mg&#47;dL&#44; a 24-h proteinuria &#40;Pu&#41; of 10&#46;4<span class="elsevierStyleHsp" style=""></span>g&#44; severe hypoalbuminemia of 1&#46;4<span class="elsevierStyleHsp" style=""></span>g&#47;dL&#44; mixed dyslipidemia as well as elevated sedimentation rate &#40;117<span class="elsevierStyleHsp" style=""></span>mm&#47;h&#41; and C-reactive protein &#40;77<span class="elsevierStyleHsp" style=""></span>mg&#47;L&#41;&#46; Urinary sediment also showed microscopic hematuria &#40;&#43;&#41;&#46; Nephrotic syndrome was considered&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">In this context&#44; occult neoplasia was excluded&#46; Viral and bacterial screening was negative&#44; as well as autoimmunity&#46; In relation to articular manifestations&#44; there were mostly peripheral joint complaints&#46; However&#44; as it was a seronegative disease &#40;rheumatoid factor and anti-citrulline protein antibodies&#41; and the patient also described low back pain for a long time&#44; this raised the hypothesis of spondyloarthritis&#44; which was confirmed &#40;bilateral radiographic sacroiliitis&#59; antigen HLA-B27 positive&#41; and a definitive diagnosis of ankylosing spondylitis &#40;AS&#41; was established&#46; Intestinal mucosal and renal biopsies revealed amyloid AA deposits &#40;amyloid A protein&#41;&#46; Concerning the latter&#44; light microscopy showed amorphous deposition in mesangium and invasion of the basement membrane &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#44; panel A&#41;&#44; and congo red evidenced amyloid deposits in glomeruli and small arteries &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#44; panel B&#41;&#46; IMF is also available &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#44; panel C&#41;&#46; Diffuse tubular atrophy was also described&#46; Additionally&#44; echocardiogram did not suggest cardiac involvement&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">Anticipating the need for institution of biological treatment&#44; all appropriate prophylactic measures were implemented &#40;the whole vaccination plan was updated&#44; influenza and antipneumococcal vaccination administered&#41; and microbiological screening was completed&#44; including screening for latent tuberculosis&#46; Due to positive tuberculin skin test and thoracic CT scan sequels&#44; started therapy for nine months with isoniazid &#40;300<span class="elsevierStyleHsp" style=""></span>mg&#47;day&#41;&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">At discharge date&#44; reference to pCr of 2<span class="elsevierStyleHsp" style=""></span>mg&#47;dL and albumin of 2&#46;5<span class="elsevierStyleHsp" style=""></span>g&#47;dL&#44; beyond the resolution of anasarca and remaining symptomatology&#46; Follow-up consultations in rheumatology&#44; internal medicine and nephrology were scheduled&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">Later&#44; given worsening renal function &#40;maximum pCr of 2&#46;9<span class="elsevierStyleHsp" style=""></span>mg&#47;dL&#41; and aggravated proteinuria&#44; which reached 20<span class="elsevierStyleHsp" style=""></span>g&#47;day &#40;with several hypoalbuminemia of 1&#46;9<span class="elsevierStyleHsp" style=""></span>g&#47;dL&#41;&#44; biological therapy with etanercept was instituted &#40;50<span class="elsevierStyleHsp" style=""></span>mg&#47;week&#41;&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">One-year follow-up showed improvement in renal function&#44; with pCr reduction from 2&#46;9 to 1&#46;6<span class="elsevierStyleHsp" style=""></span>mg&#47;dL&#44; proteinuria up to 5&#46;1<span class="elsevierStyleHsp" style=""></span>g&#47;day&#44; albuminemia from 1&#46;9<span class="elsevierStyleHsp" style=""></span>g&#47;dL to 3&#46;4<span class="elsevierStyleHsp" style=""></span>g&#47;dL and sedimentation rate from 68<span class="elsevierStyleHsp" style=""></span>mm&#47;h to 39<span class="elsevierStyleHsp" style=""></span>mm&#47;h&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">At 48 months of follow-up&#44; pCr is stable &#40;1&#46;4<span class="elsevierStyleHsp" style=""></span>mg&#47;dL&#41; and proteinuria improved&#44; under 400<span class="elsevierStyleHsp" style=""></span>mg&#47;day&#46; About AS&#44; also with a good and sustained response&#44; with low disease activity&#44; keeping etanercept as the only immunosuppressive treatment&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">Success after administration of etanercept &#8211; widely used in the treatment of inflammatory arthropathies &#8211; demonstrates once again that one of the key factors in the treatment and control of AA amyloidosis lies in controlling the underlying disease despite the presence of histologically chronic stigmas&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">3</span></a></p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Funding</span><p id="par0060" class="elsevierStylePara elsevierViewall">This research did not receive any specific grant from funding agencies in the public&#44; 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Letter to the Editor
Etanercept in the treatment of ankylosing spondylitis and nephrotic syndrome in the context of AA amyloidosis: A 48-month follow-up
Etanercept en el tratamiento de la espondilitis anquilosante y el síndrome nefrótico en el contexto de la amiloidosis aa: un seguimiento de 48 meses
Ana Domingosa,
Corresponding author
ana_t_d@sapo.pt

Corresponding author.
, Joana Vidinhaa, Rui Osóriob, Teresa Jerónimoa, Célia Ribeiroc, Catarina Mendonçab, Mário Góisd, Pedro Leão Nevesa,e
a Division of Nephrology – Centro Hospitalar e Universitário do Algarve – Faro, Portugal
b Division of Internal Medicine – Centro Hospitalar e Universitário do Algarve – Faro, Portugal
c Division of Rheumatology – Centro Hospitalar e Universitário do Algarve – Faro, Portugal
d Laboratory of Renal Morphology, Hospital de Curry Cabral, Centro Hospitalar e Universitário de Lisboa Central, Portugal
e University of Algarve – Department of Biomedical Sciences and Medicine – Faro, Portugal
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Amyloidosis represents a family of diseases characterized by the deposition of proteinaceous material in the extracellular space that&#44; by forming insoluble clusters on various tissues and organs&#44; affects its function&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">1</span></a> Amyloidosis can be classified as systemic or localized&#44; acquired or hereditary and according to their constitutive proteins&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">3</span></a> Systemic subtypes consist of primary AL amyloidosis&#44; secondary amyloid A &#40;AA&#41;&#44; familial amyloidosis and &#946;2-microglobulin-related amyloidosis&#46;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">1&#44;4</span></a> AA amyloidosis is classically associated with chronic infections &#40;essentially in developing countries&#41;&#44; inflammatory chronic diseases &#40;such as sprondyloarthropathy&#44; inflammatory bowel disease and rheumatoid arthritis&#41;&#44; periodic fever syndromes&#44; among other examples&#46;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">1&#8211;4</span></a> Clinical and laboratory manifestations depend on the type of amyloidosis and may include&#44; for example&#44; easy bruising&#44; macroglossia&#44; signs and symptoms of heart failure or arrhythmias&#44; hepatomegaly&#44; coagulation disorders or nephrotic proteinuria&#46;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">1&#8211;3</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We present a case of an older woman with nephrotic syndrome and a previously non clarified inflammatory peripheral arthropathy&#44; with the latter becoming responsible for AA amyloidosis&#46; This 48-month follow-up proves the need and effectiveness of properly treating the underlying disease&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">O&#46; G&#46; D&#46;&#44; a 78-year-old caucasian woman with a personal history of seronegative peripheral polyarthritis&#44; poor therapeutic compliance and corticoid-induced diabetes mellitus&#44; was admitted for etiological study of constitutional syndrome&#44; inflammatory arthralgias&#44; diarrhea and anasarca refractory to oral diuretics&#46; Usual medication included methotrexate&#44; deflazacorte&#44; esomeprazole and tapentadol&#44; folic acid&#44; calcium carbonate plus cholecalciferol and insulin&#46; Denied drug allergies&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">The complementary study revealed anemia &#40;normocytic&#44; normochromic&#41; of 9&#46;2<span class="elsevierStyleHsp" style=""></span>g&#47;dL&#44; normal leukogram&#44; thrombocytosis &#40;720<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>10&#94;9&#47;L&#41;&#44; pCr of 1&#46;3<span class="elsevierStyleHsp" style=""></span>mg&#47;dL&#44; a 24-h proteinuria &#40;Pu&#41; of 10&#46;4<span class="elsevierStyleHsp" style=""></span>g&#44; severe hypoalbuminemia of 1&#46;4<span class="elsevierStyleHsp" style=""></span>g&#47;dL&#44; mixed dyslipidemia as well as elevated sedimentation rate &#40;117<span class="elsevierStyleHsp" style=""></span>mm&#47;h&#41; and C-reactive protein &#40;77<span class="elsevierStyleHsp" style=""></span>mg&#47;L&#41;&#46; Urinary sediment also showed microscopic hematuria &#40;&#43;&#41;&#46; Nephrotic syndrome was considered&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">In this context&#44; occult neoplasia was excluded&#46; Viral and bacterial screening was negative&#44; as well as autoimmunity&#46; In relation to articular manifestations&#44; there were mostly peripheral joint complaints&#46; However&#44; as it was a seronegative disease &#40;rheumatoid factor and anti-citrulline protein antibodies&#41; and the patient also described low back pain for a long time&#44; this raised the hypothesis of spondyloarthritis&#44; which was confirmed &#40;bilateral radiographic sacroiliitis&#59; antigen HLA-B27 positive&#41; and a definitive diagnosis of ankylosing spondylitis &#40;AS&#41; was established&#46; Intestinal mucosal and renal biopsies revealed amyloid AA deposits &#40;amyloid A protein&#41;&#46; Concerning the latter&#44; light microscopy showed amorphous deposition in mesangium and invasion of the basement membrane &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#44; panel A&#41;&#44; and congo red evidenced amyloid deposits in glomeruli and small arteries &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#44; panel B&#41;&#46; IMF is also available &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#44; panel C&#41;&#46; Diffuse tubular atrophy was also described&#46; Additionally&#44; echocardiogram did not suggest cardiac involvement&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">Anticipating the need for institution of biological treatment&#44; all appropriate prophylactic measures were implemented &#40;the whole vaccination plan was updated&#44; influenza and antipneumococcal vaccination administered&#41; and microbiological screening was completed&#44; including screening for latent tuberculosis&#46; Due to positive tuberculin skin test and thoracic CT scan sequels&#44; started therapy for nine months with isoniazid &#40;300<span class="elsevierStyleHsp" style=""></span>mg&#47;day&#41;&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">At discharge date&#44; reference to pCr of 2<span class="elsevierStyleHsp" style=""></span>mg&#47;dL and albumin of 2&#46;5<span class="elsevierStyleHsp" style=""></span>g&#47;dL&#44; beyond the resolution of anasarca and remaining symptomatology&#46; Follow-up consultations in rheumatology&#44; internal medicine and nephrology were scheduled&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">Later&#44; given worsening renal function &#40;maximum pCr of 2&#46;9<span class="elsevierStyleHsp" style=""></span>mg&#47;dL&#41; and aggravated proteinuria&#44; which reached 20<span class="elsevierStyleHsp" style=""></span>g&#47;day &#40;with several hypoalbuminemia of 1&#46;9<span class="elsevierStyleHsp" style=""></span>g&#47;dL&#41;&#44; biological therapy with etanercept was instituted &#40;50<span class="elsevierStyleHsp" style=""></span>mg&#47;week&#41;&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">One-year follow-up showed improvement in renal function&#44; with pCr reduction from 2&#46;9 to 1&#46;6<span class="elsevierStyleHsp" style=""></span>mg&#47;dL&#44; proteinuria up to 5&#46;1<span class="elsevierStyleHsp" style=""></span>g&#47;day&#44; albuminemia from 1&#46;9<span class="elsevierStyleHsp" style=""></span>g&#47;dL to 3&#46;4<span class="elsevierStyleHsp" style=""></span>g&#47;dL and sedimentation rate from 68<span class="elsevierStyleHsp" style=""></span>mm&#47;h to 39<span class="elsevierStyleHsp" style=""></span>mm&#47;h&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">At 48 months of follow-up&#44; pCr is stable &#40;1&#46;4<span class="elsevierStyleHsp" style=""></span>mg&#47;dL&#41; and proteinuria improved&#44; under 400<span class="elsevierStyleHsp" style=""></span>mg&#47;day&#46; About AS&#44; also with a good and sustained response&#44; with low disease activity&#44; keeping etanercept as the only immunosuppressive treatment&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">Success after administration of etanercept &#8211; widely used in the treatment of inflammatory arthropathies &#8211; demonstrates once again that one of the key factors in the treatment and control of AA amyloidosis lies in controlling the underlying disease despite the presence of histologically chronic stigmas&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">3</span></a></p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Funding</span><p id="par0060" class="elsevierStylePara elsevierViewall">This research did not receive any specific grant from funding agencies in the public&#44; commercial&#44; or not-for-profit sectors&#46;</p></span></span>"
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Article information
ISSN: 20132514
Original language: English
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