Letter to the Editor
A case of relapsed anti-glomerular basement membrane glomerulonephritis complicated by IgG4-related disease
Un caso de glomerulonefritis recidivante de la membrana basal antiglomerular complicada por una enfermedad relacionada con la IgG4
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"apellidos" => "Abe" "email" => array:1 [ 0 => "a.abe@sapmed.ac.jp" ] "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" => "Yoshinosuke" "apellidos" => "Shimamura" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "Keitaro" "apellidos" => "Nishizawa" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 3 => array:3 [ "nombre" => "Takuto" "apellidos" => "Maeda" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 4 => array:3 [ "nombre" => "Kei" "apellidos" => "Yane" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 5 => array:3 [ "nombre" => "Toshiya" "apellidos" => "Shinohara" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] 6 => array:3 [ "nombre" => "Yayoi" "apellidos" => "Ogawa" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">d</span>" "identificador" => "aff0020" ] ] ] 7 => array:3 [ "nombre" => "Hideki" "apellidos" => "Takizawa" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:4 [ 0 => array:3 [ "entidad" => "Department of Nephrology, Teine Keijinkai Medical Center, Sapporo, Japan" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Department of Gastroenterology, Teine Keijinkai Medical Center, Sapporo, Japan" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Department of Pathology, Teine Keijinkai Medical Center, Sapporo, Japan" "etiqueta" => "c" "identificador" => "aff0015" ] 3 => array:3 [ "entidad" => "Hokkaido Renal Pathology Center, Sapporo, Japan" "etiqueta" => "d" "identificador" => "aff0020" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "<span class="elsevierStyleItalic">Corresponding author</span>." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Un caso de glomerulonefritis recidivante de la membrana basal antiglomerular complicada por una enfermedad relacionada con la IgG4" ] ] "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" => 992 "Ancho" => 2500 "Tamanyo" => 553058 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Histological sections of pancreatic fine-needle aspiration specimens. (A) Hematoxylin and eosin staining showing storiform fibrosis (original magnification ×400). (B) IgG4-immunohistochemical staining showing a large number of IgG4-positive plasma cells (>10 per high-power field, original magnification ×400).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Anti-glomerular basement membrane (GBM) glomerulonephritis is an autoimmune disease triggered by autoantibodies against the NC1 domain of the α3 chain of type IV collagen (α3(IV)NC1). Anti-GBM glomerulonephritis rarely relapse after remission.<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">1,2</span></a> Rare cases of anti-GBM glomerulonephritis complicated by IgG4-related disease (IgG4-RD) have been reported.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">3</span></a> Here, we report a rare case of anti-GBM glomerulonephritis complicated by type 1 autoimmune pancreatitis (AIP), a pancreatic manifestation of IgG4-RD, in a patient who experienced a relapse after remission.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Case report</span><p id="par0010" class="elsevierStylePara elsevierViewall">A 72-year-old man with fever and back pain was admitted to our institute. Three months before admission, he was diagnosed as having AIP based on the findings of pancreatic tail enlargement, irregular narrowing of the main pancreatic duct, and infiltration of lymphocytes and IgG4-positive plasma cells, together with storiform fibrosis by a pancreatic biopsy (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>), in accordance with the Japanese diagnostic criteria for AIP.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">4</span></a> He had been followed up without any medication. He had no history of smoking. His physical examination results were unremarkable except for a fever with a body temperature of 38.0<span class="elsevierStyleHsp" style=""></span>°C. Laboratory studies showed worsening of his renal function (increased serum creatinine [Cr] level to 1.77<span class="elsevierStyleHsp" style=""></span>mg/dL from a baseline of 0.5<span class="elsevierStyleHsp" style=""></span>mg/dL), proteinuria (2.07<span class="elsevierStyleHsp" style=""></span>g/gCr), hematuria (30–49 red blood cells per high-power field), and a C-reactive protein level of 30.9<span class="elsevierStyleHsp" style=""></span>mg/dL. His serological test results were positive for anti-GBM antibodies (1500<span class="elsevierStyleHsp" style=""></span>U/mL) but negative for anti-neutrophil cytoplasmic antibodies (ANCAs). Plain computed tomography revealed no signs of alveolar hemorrhage or acute pancreatitis. Renal biopsy revealed 8 glomeruli, all of which had a cellular crescent formation. No signs of IgG4-related tubulointerstitial nephritis (TIN) were found. Immunofluorescence microscopy revealed positive linear staining for IgG (IgG1<span class="elsevierStyleHsp" style=""></span>><span class="elsevierStyleHsp" style=""></span>IgG4) and C3 along the GBM (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). On the basis of these findings, the patient was diagnosed as having anti-GBM glomerulonephritis. He received intravenous pulse methylprednisolone (500<span class="elsevierStyleHsp" style=""></span>mg daily for 3 days) followed by prednisolone (30<span class="elsevierStyleHsp" style=""></span>mg daily) and plasma exchange (three times weekly for 25 occurrences). Five months later, his anti-GBM antibody level was undetectable, so the prednisolone dosage was tapered to 5<span class="elsevierStyleHsp" style=""></span>mg daily with a stable serum Cr level of 2.42<span class="elsevierStyleHsp" style=""></span>mg/dL. One year after treatment initiation, his serum Cr level increased to 7.40<span class="elsevierStyleHsp" style=""></span>mg/dL and he tested positive for anti-GBM antibodies (681<span class="elsevierStyleHsp" style=""></span>U/mL). He was diagnosed as having relapsed anti-GBM glomerulonephritis and underwent hemodialysis. He was treated with intravenous pulse methylprednisolone followed by prednisolone and plasma exchange. Nine weeks later, his anti-GBM antibody level was successfully reduced to 8.3<span class="elsevierStyleHsp" style=""></span>U/mL, but he remained dialysis dependent.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Discussion</span><p id="par0015" class="elsevierStylePara elsevierViewall">We report a rare case of relapsed anti-GBM glomerulonephritis complicated by AIP. Our case highlights two important clinical issues: anti-GBM glomerulonephritis can relapse after remission and can be complicated by IgG4-RD.</p><p id="par0020" class="elsevierStylePara elsevierViewall">In typical anti-GBM disease, the risk of relapse is thought to be quite low (1.2–2.3%).<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">1,2</span></a> Although a case of relapse triggered by smoking has been reported,<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">5</span></a> our patient had no history of smoking. Recently, two atypical variants of anti-GBM disease have been reported. One is anti-GBM disease with double seropositivity, for ANCAs and anti-GBM antibodies, with a relapse risk of 22%.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">6</span></a> The other variant is anti-GBM disease with a predominance of subclass IgG4, with a relapse risk of 50%.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">7</span></a> We ruled out these atypical types.</p><p id="par0025" class="elsevierStylePara elsevierViewall">To our knowledge, no previous reports have described cases of anti-GBM glomerulonephritis complicated by AIP. One report described two cases of anti-GBM disease complicated by IgG4-related TIN.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">3</span></a> Both cases presented with acute kidney injury and were diagnosed as anti-GBM glomerulonephritis and IgG4-related TIN based on the findings of seropositivity for anti-GBM antibodies and IgG linear deposition in the GBM and infiltration of IgG4-positive plasma cells by renal biopsy. Both cases were serologically double-positive for anti-GBM antibodies and ANCAs, and had a poor prognosis.</p><p id="par0030" class="elsevierStylePara elsevierViewall">It is important to distinguish IgG4-RD from Castleman disease (CD) because both show plasma cell infiltration. CD is characterized by lymph node proliferation and systemic inflammation. A case series study reported three cases of anti-GBM disease complicated by CD.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">8</span></a> The authors detected plasma cells secreting anti-α3(IV)NC1 antibodies in lymph nodes. One of the three patients experienced a relapse. Therefore, they suggested a causal relationship between plasma cell infiltration and anti-GBM antibody production. In our case, plasma cell infiltration was observed in the pancreas; therefore, our patient may be at high risk of relapse and require careful follow-up.</p><p id="par0035" class="elsevierStylePara elsevierViewall">In conclusion, anti-GBM glomerulonephritis can be complicated by IgG4-RD and relapse after remission.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Compliance with ethical standards</span><p id="par0040" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare. This research did not receive any specific grant from funding agencies. All procedures performed were in accordance with the ethical standards of the institutional research committee (IRB approval number: 3-019068-00) and with the Helsinki declaration. Informed consent was obtained from the patient.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Conflict of interest</span><p id="par0045" class="elsevierStylePara elsevierViewall">None.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:6 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Case report" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Discussion" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Compliance with ethical standards" ] 3 => array:2 [ "identificador" => "sec0020" "titulo" => "Conflict of interest" ] 4 => array:2 [ "identificador" => "xack529205" "titulo" => "Acknowledgements" ] 5 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "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" => 992 "Ancho" => 2500 "Tamanyo" => 553058 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Histological sections of pancreatic fine-needle aspiration specimens. (A) Hematoxylin and eosin staining showing storiform fibrosis (original magnification ×400). (B) IgG4-immunohistochemical staining showing a large number of IgG4-positive plasma cells (>10 per high-power field, original magnification ×400).</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Fig. 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 989 "Ancho" => 2500 "Tamanyo" => 632012 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Histological sections of renal biopsy specimens. (A) Periodic acid-Schiff staining showing a cellular crescent formation (original magnification ×400). (B) Immunofluorescence staining showing linear patterns of IgG deposition along the glomerular basement membrane (original magnification ×400).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:8 [ 0 => array:3 [ "identificador" => "bib0045" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Long-term outcome of anti-glomerular basement membrane antibody disease treated with plasma exchange and immunosuppression" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "J.B. Levy" 1 => "A.N. Turner" 2 => "A.J. Rees" 3 => "C.D. 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Cui" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1053/j.ajkd.2018.01.029" "Revista" => array:6 [ "tituloSerie" => "Am J Kidney Dis" "fecha" => "2018" "volumen" => "71" "paginaInicial" => "904" "paginaFinal" => "908" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/29510921" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] "agradecimientos" => array:1 [ 0 => array:4 [ "identificador" => "xack529205" "titulo" => "Acknowledgements" "texto" => "<p id="par0050" class="elsevierStylePara elsevierViewall">We would like to thank Editage (<a target="_blank" href="http://www.editage.jp/">www.editage.jp</a>) for English language editing.</p>" "vista" => "all" ] ] ] "idiomaDefecto" => "en" "url" => "/02116995/0000004100000003/v1_202105080820/S0211699520300965/v1_202105080820/en/main.assets" "Apartado" => array:4 [ "identificador" => "48186" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Cartas al Director" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/02116995/0000004100000003/v1_202105080820/S0211699520300965/v1_202105080820/en/main.pdf?idApp=UINPBA000064&text.app=https://revistanefrologia.com/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0211699520300965?idApp=UINPBA000064" ]
| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 10 | 8 | 18 |
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| 2023 Septiembre | 61 | 32 | 93 |
| 2023 Agosto | 52 | 27 | 79 |
| 2023 Julio | 64 | 22 | 86 |
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| 2023 Abril | 65 | 25 | 90 |
| 2023 Marzo | 75 | 30 | 105 |
| 2023 Febrero | 56 | 19 | 75 |
| 2023 Enero | 49 | 28 | 77 |
| 2022 Diciembre | 82 | 39 | 121 |
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| 2022 Abril | 69 | 68 | 137 |
| 2022 Marzo | 51 | 48 | 99 |
| 2022 Febrero | 71 | 56 | 127 |
| 2022 Enero | 78 | 45 | 123 |
| 2021 Diciembre | 59 | 37 | 96 |
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| 2021 Octubre | 63 | 60 | 123 |
| 2021 Septiembre | 50 | 33 | 83 |
| 2021 Agosto | 48 | 63 | 111 |
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| 2021 Marzo | 59 | 33 | 92 |
| 2021 Febrero | 27 | 21 | 48 |
| 2021 Enero | 22 | 17 | 39 |
| 2020 Diciembre | 24 | 13 | 37 |
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