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and sediment 20 RBCs&#47;field &#40;56&#37; dysmorphic&#41;&#46; Positive routine autoimmunity &#40;ANA&#43;&#44; Ro&#43;&#44; La&#43;&#44; C4 hypocomplementaemia 1<span class="elsevierStyleHsp" style=""></span>mg&#47;dl and very high IgM and rheumatoid factor &#91;RF&#93;&#41;&#46; In addition to current C3 hypocomplementaemia 66<span class="elsevierStyleHsp" style=""></span>mg&#47;dl&#44; drop in IgG 600<span class="elsevierStyleHsp" style=""></span>mg&#47;dl &#40;previously normal&#41; and positive for cryoglobulins&#46; Negative hepatitis C and B and HIV serology&#46; Negative ANCAs&#46; A skin biopsy was performed and reported small-vessel leukocytoclastic vasculitis&#46; A renal biopsy of 28 glomeruli reported the following&#58; glomerulomegaly&#59; in 3 glomeruli&#44; widespread sclerosis and&#44; in the rest of glomeruli&#44; lesions of ischaemia&#8211;basement membrane folding&#44; mesangial and endocapillary hypercellularity&#44; and some small PAS<span class="elsevierStyleHsp" style=""></span>&#43;<span class="elsevierStyleHsp" style=""></span>hyaline thrombi with inflammatory cells CD68&#43; in their interior&#59; mononuclear interstitial infiltrate&#59; mild fibrosis and atrophy&#59; focal tubular inflammation and ischaemia&#59; arterioles with focal deposition of PAS<span class="elsevierStyleHsp" style=""></span>&#43;<span class="elsevierStyleHsp" style=""></span>hyaline substance and thickening of the intima on immunofluorescence &#40;9 glomeruli&#47;1 sclerotic&#41;&#59; IgM&#43; in interstitial inflammatory cell foci and around tubules&#59; C3&#177; heterogeneous mesangial C3 and in vascular walls&#59; and negative Epstein&#8211;Barr virus <span class="elsevierStyleItalic">in situ</span> hybridisation &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Given such findings&#44; she was diagnosed with mixed cryoglobulinaemia associated with pSS&#44; and it was decided to start treatment with rituximab 375<span class="elsevierStyleHsp" style=""></span>mg&#47;m<span class="elsevierStyleSup">2</span>&#47;week &#40;4 doses&#41; and to maintain azathioprine and prednisone at routine doses&#46; The result was satisfactory&#58; CD20 lymphocytes dropped to 0&#46;3&#37;&#44; the anaemia and renal profile &#40;sediment and clearance&#41; returned to normal and she was negative for cryoglobulins&#46; At her last follow-up &#40;25 months post-biopsy&#41;&#44; the stability of renal function and percentage of CD20 B lymphocytes persisted&#44; but she was again positive for cryoglobulins at her last 2 follow-ups&#46; Now&#44; we are expectant&#44; with close monitoring&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">Notable in this case was the low clinical expressivity of the patient&#39;s cryoglobulinaemic glomerulonephritis&#44; which we attributed to early clinical diagnosis&#46; The indication for a renal biopsy was relative&#44; given the mild nature of the clinical and laboratory findings&#44; but baseline disease and the large variety of potential associated nephropathies<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> led us to perform the renal biopsy&#46; Had it been delayed&#44; she probably would have had greater parenchymal damage&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">The most commonly reported finding in kidney biopsy of pSS is tubulointerstitial disease&#59; glomerular disease is rare and coexistence of the two as in the patient described is uncertain&#46; The presence of the glomerular and cryoglobulinaemic component seems to worsen the prognosis of survival&#46;<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">1&#44;2</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Induction with Rituximab in cryoglobulinaemic glomerulonephritis produces positive results in terms of the balance efficacy&#47;safety&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> The baseline level of CD27&#43; memory lymphocytes seems to predict a better response to this drug&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a> With respect to maintenance treatment&#44; nothing has been established yet&#46; A relapse rate of up to 40&#37; has been reported&#44; but this was in series with a majority of patients with HCV&#43; and without consideration of predictive factors&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a> Reactivating the complement&#44; cryoglobulins or CD20 cells does not in itself is not an indication of a new cycle of immunosuppressant therapy&#44; but it suggest a closer follow-up as a precaution in anticipation of a clinical recurrence&#46; This is the attitude adopted in this case&#46;</p></span>"
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Letter to the Editor
Sjogren syndrome and mixed nephropathy. Significance of early kidney biopsy
Síndrome de Sjogren y nefropatía mixta. La importancia de la precocidad en la biopsia renal
M. Adoración Martín-Gómeza,
Corresponding author
doritamg@gmail.com

Corresponding author.
, Mercedes Caba Molinab, Gracia Cruz Caparrosc, Javier Muñoz Vicod, Mercedes Gómez Moralesb
a Unidad de Nefrología, Hospital de Poniente, El Ejido, Almería, Spain
b Servicio de Anatomía Patológica, Hospital San Cecilio, Granada, Spain
c Medicina Interna, Hospital de Poniente, El Ejido, Almería, Spain
d Inmunología Hospital Torrecárdenas, Almería, Spain
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weakness&#44; fever of 37&#46;8<span class="elsevierStyleHsp" style=""></span>&#176;C&#44; dry cough&#44; nausea and bilious vomiting&#44; together with weight loss of up to 10<span class="elsevierStyleHsp" style=""></span>kg in 2 months&#46; Findings on physical examination included skin purpura and bilateral basal hypoventilation&#44; attributed to her pulmonary fibrosis&#46; She was stable as confirmed by CT scan &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#44; with no palpable lymphadenopathies&#46; Findings on laboratory testing included acute normochromic normocytic anaemia that was neither haemolytic nor iron-deficient&#44; with normal protein electrophoresis&#44; creatinine 1&#46;07<span class="elsevierStyleHsp" style=""></span>mg&#47;dl &#40;reatinine clearance of 68<span class="elsevierStyleHsp" style=""></span>ml&#47;min&#41;&#44; proteinuria 200<span class="elsevierStyleHsp" style=""></span>mg&#47;dl &#40;0&#46;38<span class="elsevierStyleHsp" style=""></span>g&#47;24<span class="elsevierStyleHsp" style=""></span>h&#41; and sediment 20 RBCs&#47;field &#40;56&#37; dysmorphic&#41;&#46; Positive routine autoimmunity &#40;ANA&#43;&#44; Ro&#43;&#44; La&#43;&#44; C4 hypocomplementaemia 1<span class="elsevierStyleHsp" style=""></span>mg&#47;dl and very high IgM and rheumatoid factor &#91;RF&#93;&#41;&#46; In addition to current C3 hypocomplementaemia 66<span class="elsevierStyleHsp" style=""></span>mg&#47;dl&#44; drop in IgG 600<span class="elsevierStyleHsp" style=""></span>mg&#47;dl &#40;previously normal&#41; and positive for cryoglobulins&#46; Negative hepatitis C and B and HIV serology&#46; Negative ANCAs&#46; A skin biopsy was performed and reported small-vessel leukocytoclastic vasculitis&#46; A renal biopsy of 28 glomeruli reported the following&#58; glomerulomegaly&#59; in 3 glomeruli&#44; widespread sclerosis and&#44; in the rest of glomeruli&#44; lesions of ischaemia&#8211;basement membrane folding&#44; mesangial and endocapillary hypercellularity&#44; and some small PAS<span class="elsevierStyleHsp" style=""></span>&#43;<span class="elsevierStyleHsp" style=""></span>hyaline thrombi with inflammatory cells CD68&#43; in their interior&#59; mononuclear interstitial infiltrate&#59; mild fibrosis and atrophy&#59; focal tubular inflammation and ischaemia&#59; arterioles with focal deposition of PAS<span class="elsevierStyleHsp" style=""></span>&#43;<span class="elsevierStyleHsp" style=""></span>hyaline substance and thickening of the intima on immunofluorescence &#40;9 glomeruli&#47;1 sclerotic&#41;&#59; IgM&#43; in interstitial inflammatory cell foci and around tubules&#59; C3&#177; heterogeneous mesangial C3 and in vascular walls&#59; and negative Epstein&#8211;Barr virus <span class="elsevierStyleItalic">in situ</span> hybridisation &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Given such findings&#44; she was diagnosed with mixed cryoglobulinaemia associated with pSS&#44; and it was decided to start treatment with rituximab 375<span class="elsevierStyleHsp" style=""></span>mg&#47;m<span class="elsevierStyleSup">2</span>&#47;week &#40;4 doses&#41; and to maintain azathioprine and prednisone at routine doses&#46; The result was satisfactory&#58; CD20 lymphocytes dropped to 0&#46;3&#37;&#44; the anaemia and renal profile &#40;sediment and clearance&#41; returned to normal and she was negative for cryoglobulins&#46; At her last follow-up &#40;25 months post-biopsy&#41;&#44; the stability of renal function and percentage of CD20 B lymphocytes persisted&#44; but she was again positive for cryoglobulins at her last 2 follow-ups&#46; Now&#44; we are expectant&#44; with close monitoring&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">Notable in this case was the low clinical expressivity of the patient&#39;s cryoglobulinaemic glomerulonephritis&#44; which we attributed to early clinical diagnosis&#46; The indication for a renal biopsy was relative&#44; given the mild nature of the clinical and laboratory findings&#44; but baseline disease and the large variety of potential associated nephropathies<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> led us to perform the renal biopsy&#46; Had it been delayed&#44; she probably would have had greater parenchymal damage&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">The most commonly reported finding in kidney biopsy of pSS is tubulointerstitial disease&#59; glomerular disease is rare and coexistence of the two as in the patient described is uncertain&#46; The presence of the glomerular and cryoglobulinaemic component seems to worsen the prognosis of survival&#46;<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">1&#44;2</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Induction with Rituximab in cryoglobulinaemic glomerulonephritis produces positive results in terms of the balance efficacy&#47;safety&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> The baseline level of CD27&#43; memory lymphocytes seems to predict a better response to this drug&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a> With respect to maintenance treatment&#44; nothing has been established yet&#46; A relapse rate of up to 40&#37; has been reported&#44; but this was in series with a majority of patients with HCV&#43; and without consideration of predictive factors&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a> Reactivating the complement&#44; cryoglobulins or CD20 cells does not in itself is not an indication of a new cycle of immunosuppressant therapy&#44; but it suggest a closer follow-up as a precaution in anticipation of a clinical recurrence&#46; This is the attitude adopted in this case&#46;</p></span>"
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ISSN: 20132514
Original language: English
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Nefrología (English Edition)