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Biopsy of the auricular cartilage was performed&#44; which confirmed the diagnosis of RP&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">In the laboratory tests&#44; the patient presented mild normocytic anaemia&#44; leucocytosis&#44; thrombocytosis and elevation of acute-phase reactants &#40;C-reactive protein 22<span class="elsevierStyleHsp" style=""></span>mg&#47;dl&#41;&#44; with sterile blood cultures and urine culture&#46; Kidney function was normal&#44; with proteinuria of 0&#46;5<span class="elsevierStyleHsp" style=""></span>g&#47;24<span class="elsevierStyleHsp" style=""></span>h and microscopic haematuria&#46; Antinuclear antibodies and anti-DNA and the rheumatoid factor were negative&#44; complement &#40;C3 and C4&#41; values were in the normal range on several occasions&#46; Kidney function was maintained within normal limits&#44; but the patient presented proteinuria of 2&#46;9<span class="elsevierStyleHsp" style=""></span>g&#47;24<span class="elsevierStyleHsp" style=""></span>h and abnormalities in the urinary sediment which included microscopic haematuria and leucocyturia&#46; This is why a kidney biopsy was performed&#46; Optical microscopy showed findings compatible with focal segmental hyalinosis with periglomerular sclerosis &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; Segmental IgM deposits were revealed using the immunofluorescence technique&#44; and an irregular podocyte effacement was observed using electron microscopy &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; Treatment was started with prednisone at 1<span class="elsevierStyleHsp" style=""></span>mg&#47;kg weight and captopril at maximum doses&#44; with a favourable clinical course of the patient&#39;s polychondritis and renal response with reduction of proteinuria to 0&#46;4<span class="elsevierStyleHsp" style=""></span>g&#47;24<span class="elsevierStyleHsp" style=""></span>h and disappearance of microscopic haematuria&#46; After discharge from Nephrology&#44; steroids were maintained for six months due to his RP and steroid treatment was subsequently received on request for short periods&#44; according to the joint inflammation symptoms&#46; During his clinical course&#44; at points the patient tested positive for cytoplasmic antineutrophil cytoplasmic antibodies &#40;c-ANCA&#41; against serine proteinase-3 &#40;PR3&#41; at low titres&#44; but without presenting clinical data of vasculitis or other extrarenal manifestations&#46; Anti-myeloperoxidase &#40;MPO&#41; antibodies and cryoglobulins were negative&#46; After 32 years of evolution of his condition&#44; the patient has presented acceptable progress of his kidney function &#40;serum creatinine of 1&#46;4<span class="elsevierStyleHsp" style=""></span>mg&#47;dl and proteinuria of 0&#46;6<span class="elsevierStyleHsp" style=""></span>g&#47;24<span class="elsevierStyleHsp" style=""></span>h&#41; on treatment with renin&#8211;angiotensin axis blocking drugs&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">RP is a rare autoimmune disease which&#44; in addition to compromising cartilage structures&#44; also affects non-cartilage structures with a high content of glycosaminoglycans&#44; such as the lungs&#44; heart&#44; eyes and blood vessels&#46; Renal involvement in the clinical course of RP is very rare and it was Neil et al&#46;&#44; in 1978&#44; who reported the first cases of glomerular disease associated with RP&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">2</span></a> In the literature review&#44; the prevalence of renal involvement in RP is highly variable&#46; It is possible to find historical series such as that of the Mayo Clinic&#44; with a prevalence of 22&#37; renal involvement according to clinical criteria &#40;presence of haematuria or proteinuria&#41; or histological criteria &#40;with the most prevalent lesions being mesangial nephropathy and segmental necrotising and crescentic glomerulonephritis&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">1</span></a> However&#44; more recent studies report a significantly lower percentage than that reported previously&#46; In a German series&#44; which included 62 patients with RP&#44; only 6&#46;5&#37; had associated renal damage&#44;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">3</span></a> and in another large Asian study which evaluated the clinical and prognostic characteristics of 158 patients with RP&#44; the prevalence of renal involvement was only 3&#37;&#46;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">4</span></a> In a recent review&#44; Dion et al&#46; analysed 142 patients with RP and no patient presented renal involvement&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">5</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">Although most of the wide range of histological lesions associated with RP which have been reported up to now correspond with segmental necrotising and crescentic glomerulonephritis&#44;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">6&#8211;8</span></a> other types of lesions have also been reported&#44; such as mesangial IgA nephropathy&#44; minimal change disease and membranous glomerulonephritis&#46;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">6&#44;9</span></a> There is considerable lack of knowledge with regard to the aetiopathogenesis of renal damage associated with RP&#44; with a possible autoimmune mechanism gaining increasing importance&#46; For this reason&#44; treatment is empirical and is based on the use of corticosteroids or other immunosuppressants&#44; according to the severity of the disease&#46; In cases of greater renal aggressiveness &#40;necrotising and crescentic glomerulonephritis&#41;&#44; the use of cyclophosphamide and plasmapheresis&#44; in addition to corticosteroids&#44; has been reported&#46;<a class="elsevierStyleCrossRefs" href="#bib0090"><span class="elsevierStyleSup">7&#44;10</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">The presence of ANCA has been reported in a percentage of cases of RP&#59; however&#44; there is diverging information with regard to the aetiopathogenic role of these in the association with vasculitis&#46; Papo et al&#46;&#44; in a series of 23 patients with RP&#44; reported ANCA positivity by immunofluorescence in 30&#37; of the cases &#40;three cases were c-ANCA and five were p-ANCA&#41;&#59; however&#44; when analysing these same patients with another more specific technique &#40;ELISA&#41;&#44; none of those who had c-ANCA were PR3-positive and four of the five p-ANCA patients were MPO-positive&#46;<a class="elsevierStyleCrossRef" href="#bib0110"><span class="elsevierStyleSup">11</span></a> Therefore&#44; although ANCA can be detected in RP&#44; its relevance requires an additional analysis in the clinical context of each patient&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Unlike other reported cases&#44; the clinical course of the kidney disease in our patient has been very favourable&#44; with the administration of corticosteroids and renin&#8211;angiotensin axis blocking drugs also coinciding with long periods of remission of his RP&#46;</p></span>"
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Letter to the Editor
Relapsing polychondritis and focal segmental glomerulosclerosis: Coincidence or causality
Policondritis recidivante y glomeruloesclerosis segmentaria y focal: coincidencia o causalidad
Elizabeth Canllavia, Marina Alonsob, Maria Fernándeza, Eduardo Gutiérreza, Enrique Moralesa,
Corresponding author
emoralesr@senefro.org

Corresponding author.
a Servicio de Nefrología, Hospital Universitario 12 de Octubre, Madrid, Spain
b Servicio de Anatomía Patológica, Hospital Universitario 12 de Octubre, Madrid, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Relapsing polychondritis &#40;RP&#41; is a rare autoimmune condition of unknown aetiology which is characterised by recurrent inflammatory flare-ups of cartilage structures mainly affecting the nasal&#44; auricular and laryngotracheal cartilage&#46; Renal involvement is unusual and&#44; when it does present&#44; it is associated with poorer survival&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">1</span></a> Below we describe a case of a patient with RP and associated glomerular disease&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 39-year-old male&#44; who was admitted to hospital in June 1986 due to fever and symmetric migratory polyarthritis of large and small joints&#44; conductive hearing loss secondary to seromucinous otitis media and conjunctivitis&#46; During the admission&#44; he presented with bilateral auricular chondritis with erythema and pain on palpitation&#46; Biopsy of the auricular cartilage was performed&#44; which confirmed the diagnosis of RP&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">In the laboratory tests&#44; the patient presented mild normocytic anaemia&#44; leucocytosis&#44; thrombocytosis and elevation of acute-phase reactants &#40;C-reactive protein 22<span class="elsevierStyleHsp" style=""></span>mg&#47;dl&#41;&#44; with sterile blood cultures and urine culture&#46; Kidney function was normal&#44; with proteinuria of 0&#46;5<span class="elsevierStyleHsp" style=""></span>g&#47;24<span class="elsevierStyleHsp" style=""></span>h and microscopic haematuria&#46; Antinuclear antibodies and anti-DNA and the rheumatoid factor were negative&#44; complement &#40;C3 and C4&#41; values were in the normal range on several occasions&#46; Kidney function was maintained within normal limits&#44; but the patient presented proteinuria of 2&#46;9<span class="elsevierStyleHsp" style=""></span>g&#47;24<span class="elsevierStyleHsp" style=""></span>h and abnormalities in the urinary sediment which included microscopic haematuria and leucocyturia&#46; This is why a kidney biopsy was performed&#46; Optical microscopy showed findings compatible with focal segmental hyalinosis with periglomerular sclerosis &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; Segmental IgM deposits were revealed using the immunofluorescence technique&#44; and an irregular podocyte effacement was observed using electron microscopy &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; Treatment was started with prednisone at 1<span class="elsevierStyleHsp" style=""></span>mg&#47;kg weight and captopril at maximum doses&#44; with a favourable clinical course of the patient&#39;s polychondritis and renal response with reduction of proteinuria to 0&#46;4<span class="elsevierStyleHsp" style=""></span>g&#47;24<span class="elsevierStyleHsp" style=""></span>h and disappearance of microscopic haematuria&#46; After discharge from Nephrology&#44; steroids were maintained for six months due to his RP and steroid treatment was subsequently received on request for short periods&#44; according to the joint inflammation symptoms&#46; During his clinical course&#44; at points the patient tested positive for cytoplasmic antineutrophil cytoplasmic antibodies &#40;c-ANCA&#41; against serine proteinase-3 &#40;PR3&#41; at low titres&#44; but without presenting clinical data of vasculitis or other extrarenal manifestations&#46; Anti-myeloperoxidase &#40;MPO&#41; antibodies and cryoglobulins were negative&#46; After 32 years of evolution of his condition&#44; the patient has presented acceptable progress of his kidney function &#40;serum creatinine of 1&#46;4<span class="elsevierStyleHsp" style=""></span>mg&#47;dl and proteinuria of 0&#46;6<span class="elsevierStyleHsp" style=""></span>g&#47;24<span class="elsevierStyleHsp" style=""></span>h&#41; on treatment with renin&#8211;angiotensin axis blocking drugs&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">RP is a rare autoimmune disease which&#44; in addition to compromising cartilage structures&#44; also affects non-cartilage structures with a high content of glycosaminoglycans&#44; such as the lungs&#44; heart&#44; eyes and blood vessels&#46; Renal involvement in the clinical course of RP is very rare and it was Neil et al&#46;&#44; in 1978&#44; who reported the first cases of glomerular disease associated with RP&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">2</span></a> In the literature review&#44; the prevalence of renal involvement in RP is highly variable&#46; It is possible to find historical series such as that of the Mayo Clinic&#44; with a prevalence of 22&#37; renal involvement according to clinical criteria &#40;presence of haematuria or proteinuria&#41; or histological criteria &#40;with the most prevalent lesions being mesangial nephropathy and segmental necrotising and crescentic glomerulonephritis&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">1</span></a> However&#44; more recent studies report a significantly lower percentage than that reported previously&#46; In a German series&#44; which included 62 patients with RP&#44; only 6&#46;5&#37; had associated renal damage&#44;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">3</span></a> and in another large Asian study which evaluated the clinical and prognostic characteristics of 158 patients with RP&#44; the prevalence of renal involvement was only 3&#37;&#46;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">4</span></a> In a recent review&#44; Dion et al&#46; analysed 142 patients with RP and no patient presented renal involvement&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">5</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">Although most of the wide range of histological lesions associated with RP which have been reported up to now correspond with segmental necrotising and crescentic glomerulonephritis&#44;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">6&#8211;8</span></a> other types of lesions have also been reported&#44; such as mesangial IgA nephropathy&#44; minimal change disease and membranous glomerulonephritis&#46;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">6&#44;9</span></a> There is considerable lack of knowledge with regard to the aetiopathogenesis of renal damage associated with RP&#44; with a possible autoimmune mechanism gaining increasing importance&#46; For this reason&#44; treatment is empirical and is based on the use of corticosteroids or other immunosuppressants&#44; according to the severity of the disease&#46; In cases of greater renal aggressiveness &#40;necrotising and crescentic glomerulonephritis&#41;&#44; the use of cyclophosphamide and plasmapheresis&#44; in addition to corticosteroids&#44; has been reported&#46;<a class="elsevierStyleCrossRefs" href="#bib0090"><span class="elsevierStyleSup">7&#44;10</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">The presence of ANCA has been reported in a percentage of cases of RP&#59; however&#44; there is diverging information with regard to the aetiopathogenic role of these in the association with vasculitis&#46; Papo et al&#46;&#44; in a series of 23 patients with RP&#44; reported ANCA positivity by immunofluorescence in 30&#37; of the cases &#40;three cases were c-ANCA and five were p-ANCA&#41;&#59; however&#44; when analysing these same patients with another more specific technique &#40;ELISA&#41;&#44; none of those who had c-ANCA were PR3-positive and four of the five p-ANCA patients were MPO-positive&#46;<a class="elsevierStyleCrossRef" href="#bib0110"><span class="elsevierStyleSup">11</span></a> Therefore&#44; although ANCA can be detected in RP&#44; its relevance requires an additional analysis in the clinical context of each patient&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Unlike other reported cases&#44; the clinical course of the kidney disease in our patient has been very favourable&#44; with the administration of corticosteroids and renin&#8211;angiotensin axis blocking drugs also coinciding with long periods of remission of his RP&#46;</p></span>"
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