Letters to the Editor - Brief comments on case reports
Membranoproliferative glomerulonephritis in a puerperal with Sjögren's syndrome: Rare finding or something else?
Glomerulonefritis membranoproliferativa en una puerpéra con síndrome de Sjögren: hallazgo raro o algo más?
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"tieneTextoCompleto" => true "saludo" => "Dear Editor," "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "190" "paginaFinal" => "191" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Rita Leal, Luís Escada, Marta Neves, Ana Galvão, Luís Freitas, Jorge Pratas, Carol Marinho, Vítor Sousa, Lígia Prado e Castro, Mário Campos" "autores" => array:10 [ 0 => array:4 [ "nombre" => "Rita" "apellidos" => "Leal" "email" => array:1 [ 0 => "rita.gcleal@gmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "Luís" "apellidos" => "Escada" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "Marta" "apellidos" => "Neves" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 3 => array:3 [ "nombre" => "Ana" "apellidos" => "Galvão" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 4 => array:3 [ "nombre" => "Luís" "apellidos" => "Freitas" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 5 => array:3 [ "nombre" => "Jorge" "apellidos" => "Pratas" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 6 => array:3 [ "nombre" => "Carol" "apellidos" => "Marinho" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 7 => array:3 [ "nombre" => "Vítor" "apellidos" => "Sousa" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 8 => array:3 [ "nombre" => "Lígia Prado e" "apellidos" => "Castro" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 9 => array:3 [ "nombre" => "Mário" "apellidos" => "Campos" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Nephrology Department, Centro Hospitalar e Universitário de Coimbra, Portugal" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Pathology Department, Centro Hospitalar e Universitário de Coimbra, Portugal" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "<span class="elsevierStyleItalic">Corresponding author</span>." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Glomerulonefritis membranoproliferativa en una puerpéra con síndrome de Sjögren: hallazgo raro o algo más?" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 713 "Ancho" => 972 "Tamanyo" => 139122 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Renal biopsy (methenamine silver stain) 200×.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">We report on a case of a 35-year-old Caucasian woman with primary Sjögren's Syndrome (pSS) diagnosed by salivary gland biopsy and IgM κ monoclonal gammopathy of undetermined significance (MGUS), treated with hydroxychloroquine 200<span class="elsevierStyleHsp" style=""></span>mg daily since she was 22 year-old.</p><p id="par0010" class="elsevierStylePara elsevierViewall">After 24 weeks into her first pregnancy, she developed marked asthenia with muscle pain and arthralgia, hypertension, oedema and palpable purpura of the lower limbs. Laboratorial testing revealed an increase in serum creatinine (sCr) (0.75<span class="elsevierStyleHsp" style=""></span>→<span class="elsevierStyleHsp" style=""></span>1.18<span class="elsevierStyleHsp" style=""></span>mg/dL), anaemia (haemoglobin (Hb)<span class="elsevierStyleHsp" style=""></span>=<span class="elsevierStyleHsp" style=""></span>8.6<span class="elsevierStyleHsp" style=""></span>g/dL), active urinary sediment and nephrotic-range proteinuria (3.8<span class="elsevierStyleHsp" style=""></span>g/24<span class="elsevierStyleHsp" style=""></span>h). She was diagnosed with preeclampsia with foetal distress and underwent a caesarean at 29 weeks of pregnancy. One week post-partum the patient was discharged home with sCr<span class="elsevierStyleHsp" style=""></span>=<span class="elsevierStyleHsp" style=""></span>0.84<span class="elsevierStyleHsp" style=""></span>mg/dL, Hb<span class="elsevierStyleHsp" style=""></span>=<span class="elsevierStyleHsp" style=""></span>12<span class="elsevierStyleHsp" style=""></span>g/dL, 24-hour proteinuria<span class="elsevierStyleHsp" style=""></span>=<span class="elsevierStyleHsp" style=""></span>1.4<span class="elsevierStyleHsp" style=""></span>g and normotensive under nifedipine 30<span class="elsevierStyleHsp" style=""></span>mg daily.</p><p id="par0015" class="elsevierStylePara elsevierViewall">Three months post-partum, she was admitted to the emergency room presenting acute pulmonary oedema requiring invasive ventilatory support. Pulmonary auscultation showed fine crackles over both lung bases, she had moderate peripheral oedema and after urinary catheterization, oligoanuria was confirmed.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Laboratory tests revealed sCr<span class="elsevierStyleHsp" style=""></span>=<span class="elsevierStyleHsp" style=""></span>4.39<span class="elsevierStyleHsp" style=""></span>mg/dL, K<span class="elsevierStyleSup">+</span><span class="elsevierStyleHsp" style=""></span>=<span class="elsevierStyleHsp" style=""></span>6.6<span class="elsevierStyleHsp" style=""></span>mmol/L, brain natriuretic peptide<span class="elsevierStyleHsp" style=""></span>=<span class="elsevierStyleHsp" style=""></span>5013<span class="elsevierStyleHsp" style=""></span>pg/mL, Hb<span class="elsevierStyleHsp" style=""></span>=<span class="elsevierStyleHsp" style=""></span>8.1<span class="elsevierStyleHsp" style=""></span>g/dL, platelet count<span class="elsevierStyleHsp" style=""></span>=<span class="elsevierStyleHsp" style=""></span>248x10<span class="elsevierStyleSup">9</span>/L and negative blood cultures. Urinalysis showed 3+ proteinuria on a dipstick test. On echocardiogram there were no signs of endocarditis and she had a ventricular ejection fraction of 50%, renal ultrasound revealed normal kidneys and chest X-ray showed large bilateral pleural effusion.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Continuous haemodiafiltration was initiated and the patient was admitted to the intensive care unit, until she gained ventilatory autonomy and suspended haemodiafiltration, being transferred to the Nephrology Department three days later for continued care.</p><p id="par0030" class="elsevierStylePara elsevierViewall">A kidney biopsy was performed revealing type I membranoproliferative glomerulonephritis with IgM (++), IgG (+) and C3 (++) deposits, glomerular capillary endotheliosis, focal and segmental thrombotic microangiopathy, tubulointerstitial nephritis and injuries of focal and discrete vasculitis. (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0035" class="elsevierStylePara elsevierViewall">Serologies for human immunodeficiency virus, hepatitis B virus and hepatitis C virus (HCV) were negative, as well as HCV RNA testing. Complement study showed low C4 (C4<0,01) and normal C3 (C3=1,36) and high rheumatoid factor (195<span class="elsevierStyleHsp" style=""></span>IU/mL; <span class="elsevierStyleItalic">N</span><span class="elsevierStyleHsp" style=""></span>=<span class="elsevierStyleHsp" style=""></span>). Cryoglobulins were positive with polyclonal IgG and monoclonal IgM and kappa light chains.</p><p id="par0040" class="elsevierStylePara elsevierViewall">The patient was treated with 1<span class="elsevierStyleHsp" style=""></span>g methylprednisolone pulses on three consecutive days and subsequent oral prednisolone at a dosage of 1<span class="elsevierStyleHsp" style=""></span>mg/kg/day.</p><p id="par0045" class="elsevierStylePara elsevierViewall">One year has passed and the patient has been weaned off steroid therapy maintaining clinical stability. Analytically, the values of sCr have stabilized at 1.0<span class="elsevierStyleHsp" style=""></span>mg/dL, with proteinuria of 150<span class="elsevierStyleHsp" style=""></span>mg/24<span class="elsevierStyleHsp" style=""></span>h (<a class="elsevierStyleCrossRef" href="#tbl0005">Table 1</a>).</p><elsevierMultimedia ident="tbl0005"></elsevierMultimedia><p id="par0050" class="elsevierStylePara elsevierViewall">The histological findings of our patients’ kidney biopsy revealed distinct injury patterns that could be inserted in various clinical pictures: glomerular capillary endotheliosis and focal and segmental thrombotic microangiopathy were expected in the context of previous preeclampsia<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> while tubulointerstitial nephritis with injuries of vasculitis were compatible with pSS.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Regarding the immune complex mediated MPGN, which might have been the cause of acute kidney injury, the differential diagnosis of its underlying cause was challenging and included autoimmune diseases, chronic infection and monoclonal gammopathy. MPGN associated with autoimmune diseases is rarely seen in patients with SS and we also excluded chronic infection. Considering monoclonal gammopathies, although the immunofluorescence microscopy on renal biopsy was not typical, our patient was diagnosed with IgM κ MGUS.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">A growing number of pathologic renal conditions are being attributed to a clonal plasma cell disorder that is less myeloma-like and more MGUS-like in terms of its bulk and proliferative rate and the term monoclonal gammopathy of renal significance (MGRS) was proposed to clarify the differences in respect to therapy.<a class="elsevierStyleCrossRefs" href="#bib0070"><span class="elsevierStyleSup">4,5</span></a> One of the conditions associated with MGRS is type II cryoglobulinaemia and there are recent reviews that show a close association between monoclonal gammopathy, cryoglobulinaemias and SS.<a class="elsevierStyleCrossRefs" href="#bib0080"><span class="elsevierStyleSup">6,7</span></a> In fact, in patients with SS and MGUS, mixed cryoglobulinaemia should always be investigated, especially if the monoclonal band is IgM k, as in the case we report.</p><p id="par0065" class="elsevierStylePara elsevierViewall">In retrospect, there was several data favouring the diagnosis of cryoglobulinaemia in association with pSS, since our patient presented with Metlzer's triad and had laboratory findings compatible with this pathology.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">In summary, our patient had a MGRS associated with type II cryoglobulinaemia, presenting with MPGN, superimposed on SS.</p><p id="par0075" class="elsevierStylePara elsevierViewall">Regarding treatment there are still many doubts about the best approach. Our patient had several poor prognostic indicators: a severe form of pSS and type II cryoglobulinaemia, both related to a higher propensity to develop a lymphoproliferative disorder, and MGUS, nowadays considered as a key marker of pSS activity.<a class="elsevierStyleCrossRefs" href="#bib0095"><span class="elsevierStyleSup">9,6,10</span></a> Considering this data, she could be a candidate for aggressive therapy with biological agents against B-cells.</p><p id="par0080" class="elsevierStylePara elsevierViewall">For now, and after suspending steroid treatment, our patient has normal kidney function without significant proteinuria or complaints. Our approach is to maintain integrated inter-department follow up and in the future, rituximab will be present as a therapeutic option.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2015-02-06" "fechaAceptado" => "2015-02-10" "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 713 "Ancho" => 972 "Tamanyo" => 139122 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Renal biopsy (methenamine silver stain) 200×.</p>" ] ] 1 => array:8 [ "identificador" => "tbl0005" "etiqueta" => "Table 1" "tipo" => "MULTIMEDIATABLA" "mostrarFloat" => true "mostrarDisplay" => false "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at1" "detalle" => "Table " "rol" => "short" ] ] "tabla" => array:1 [ "tablatextoimagen" => array:1 [ 0 => array:2 [ "tabla" => array:1 [ 0 => """ <table border="0" frame="\n \t\t\t\t\tvoid\n \t\t\t\t" class=""><thead title="thead"><tr title="table-row"><th class="td" title="table-head " align="" valign="top" scope="col" style="border-bottom: 2px solid black"> \t\t\t\t\t\t\n \t\t\t\t</th><th class="td" title="table-head " align="center" valign="top" scope="col" style="border-bottom: 2px solid black">Before pregnancy \t\t\t\t\t\t\n \t\t\t\t</th><th class="td" title="table-head " align="center" valign="top" scope="col" style="border-bottom: 2px solid black">24–29 weeks pregnancy \t\t\t\t\t\t\n \t\t\t\t</th><th class="td" title="table-head " align="center" valign="top" scope="col" style="border-bottom: 2px solid black">1<span class="elsevierStyleHsp" style=""></span>W post-partum \t\t\t\t\t\t\n \t\t\t\t</th><th class="td" title="table-head " align="center" valign="top" scope="col" style="border-bottom: 2px solid black">3<span class="elsevierStyleHsp" style=""></span>M post-partum \t\t\t\t\t\t\n \t\t\t\t</th><th class="td" title="table-head " align="center" valign="top" scope="col" style="border-bottom: 2px solid black">6<span class="elsevierStyleHsp" style=""></span>M on steroids \t\t\t\t\t\t\n \t\t\t\t</th></tr></thead><tbody title="tbody"><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">sCr \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">0.7 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">1.18 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">0.84 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">4.39 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">1.0 \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Uprot \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">0 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">3.8 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">1.4 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">2.5 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">0.3 \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">sAlb \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">4.3 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">2.9 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">3.2 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">3.2 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">4.1 \t\t\t\t\t\t\n \t\t\t\t</td></tr></tbody></table> """ ] "imagenFichero" => array:1 [ 0 => "xTab1383032.png" ] ] ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Analytical evolution of serum creatinine (sCr), proteinuria (Uprot) and serum albumin (sAlb). W<span class="elsevierStyleHsp" style=""></span>=<span class="elsevierStyleHsp" style=""></span>week; M<span class="elsevierStyleHsp" style=""></span>=<span class="elsevierStyleHsp" style=""></span>month, W<span class="elsevierStyleHsp" style=""></span>=<span class="elsevierStyleHsp" style=""></span>week.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0055" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "The glomerular injury of preeclamsia" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "I. Stillman" 1 => "S. 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