Letter to the Editor
Microscopic polyangiitis in a patient with primary biliary cirrhosis: Treatment complications
Poliangeítis microscópica en paciente con cirrosis biliar primaria. Complicaciones del tratamiento
Elena Ruiz-Ferreras
, Alberto Martín-Arribas, Guadalupe Tabernero-Fernández, José Luis Lerma-Márquez, Pilar Fraile-Gómez, Pedro García-Cosmes, José Matías Tabernero-Romo
Corresponding author
Servicio de Nefrología, Hospital Universitario de Salamanca, Salamanca, Spain
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García-Cosmes, José Matías Tabernero-Romo" "autores" => array:7 [ 0 => array:4 [ "nombre" => "Elena" "apellidos" => "Ruiz-Ferreras" "email" => array:1 [ 0 => "eruiz_burg@hotmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "Alberto" "apellidos" => "Martín-Arribas" ] 2 => array:2 [ "nombre" => "Guadalupe" "apellidos" => "Tabernero-Fernández" ] 3 => array:2 [ "nombre" => "José Luis" "apellidos" => "Lerma-Márquez" ] 4 => array:2 [ "nombre" => "Pilar" "apellidos" => "Fraile-Gómez" ] 5 => array:2 [ "nombre" => "Pedro" "apellidos" => "García-Cosmes" ] 6 => array:2 [ "nombre" => "José Matías" "apellidos" => "Tabernero-Romo" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Nefrología, Hospital Universitario de Salamanca, Salamanca, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "<span class="elsevierStyleItalic">Corresponding author</span>." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Poliangeítis microscópica en paciente con cirrosis biliar primaria. Complicaciones del tratamiento" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0010" "etiqueta" => "Fig. 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 780 "Ancho" => 996 "Tamanyo" => 97065 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Colonoscopy.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">A 70-year-old woman with a prior history of primary biliary cirrhosis diagnosed 18 months earlier, being treated with ursodeoxycholic acid 600<span class="elsevierStyleHsp" style=""></span>mg/day. No prior history of hypertension or diabetes mellitus. No known kidney diseases. Admitted to the nephrology department owing to acute renal function deterioration (creatinine: 4.12<span class="elsevierStyleHsp" style=""></span>mg/dl; urea: 122<span class="elsevierStyleHsp" style=""></span>mg/dl). Reported wasting syndrome, over a period of several months. Diuresis was maintained, and vital signs (blood pressure [BP]: 125/75<span class="elsevierStyleHsp" style=""></span>mmHg) and physical examination were normal.</p><p id="par0010" class="elsevierStylePara elsevierViewall">Findings included normocytic normochromic anaemia (Hb 10.3<span class="elsevierStyleHsp" style=""></span>g/dl), which required initiation of erythropoietin treatment. Proteinuria of 0.840<span class="elsevierStyleHsp" style=""></span>g/day with granular casts in the sediment, and ++++ microhaematuria. She tested positive for p-ANCAs at a titre of 29.1<span class="elsevierStyleHsp" style=""></span>IU/ml. She also tested positive for anti-mitochondrial antibodies that were, at stable levels, given her already known biliary cirrhosis. Considering her acute kidney failure with positive autoimmunity for p-ANCAs, she underwent a kidney biopsy, which was consistent with diffuse extracapillary proliferative glomerulonephritis with cellular crescents in 75% of her glomeruli. She was diagnosed with rapidly progressive kidney failure secondary to type <span class="elsevierStyleSmallCaps">III</span> or pauci-immune extracapillary proliferative glomerulonephritis, consistent with microscopic polyangiitis (MPA). Treatment with corticosteroids and oral cyclophosphamide (1.5<span class="elsevierStyleHsp" style=""></span>mg/kg/day) was indicated. Good response to treatment with modest kidney improvement up to a creatinine level of 3.63<span class="elsevierStyleHsp" style=""></span>mg/dl at discharge from the hospital.</p><p id="par0015" class="elsevierStylePara elsevierViewall">During subsequent follow-up in a nephrology visit, the patient was found to be asymptomatic and with a good control of BP. Her serum creatinine values decreased (1.56<span class="elsevierStyleHsp" style=""></span>mg/dl), her p-ANCA titre were also reduced (8.5<span class="elsevierStyleHsp" style=""></span>IU/ml), her proteinuria decreased (0.639<span class="elsevierStyleHsp" style=""></span>g/day) and her microhaematuria disappeared (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). In addition, her haemoglobin levels improved so erythropoietin treatment was discontinued.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Two months after hospital discharge, she was admitted to the GI department due to gastrointestinal bleeding that affected her laboratory results with an acute exacerbation of renal failure due to hypotension and volume depletion. Gastroscopy and a colonoscopy were performed; denudation with capillary bleeding upon contact plus superficial and deep ulcers were observed, thus signs of vasculitis and inflammatory bowel disease were ruled out. As for infection, she tested positive for IgMs for cytomegalovirus, and herpes virus was isolated in oropharyngeal lesions. Despite the improvement in renal function up to baseline levels, the patient had a poor clinical course, as frequent fresh bloody stools persisted and she required transfusions every 24–48<span class="elsevierStyleHsp" style=""></span>h that required admission to ICU. A repeat colonoscopy was performed, which showed multiple deep ulcers on punch biopsy with abundant bleeding (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>), and CMV of 27,784<span class="elsevierStyleHsp" style=""></span>cop/ml was confirmed with PCR. Anti-viral treatment with ganciclovir and aciclovir was administered. A diagnosis was made of lower gastrointestinal bleeding secondary to enteritis due to cytomegalovirus in an immunosuppressed patient; in addition patient had herpes virus together with haemorrhagic colitis due to direct cyclophosphamide toxicity with secondary thrombocytopenia. Patient had uncontrollable gastrointestinal bleeding and despite a reasonable response of the kidney disease, she died a month and a half after hospital admission.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">MPA is characterised by a constant and early presence of rapidly progressive glomerulonephritis. It is important to bear in mind treatment-related complications due to both direct toxicity and immunosuppression-related toxicities. In our patient's case, although her kidney disease responded well to treatment, there was a fatal complication, cytomegalovirus enteritis.<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">1,2</span></a> This led to uncontrollable lower gastrointestinal bleeding and, finally, to her death.<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">3,4</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">It is not uncommon to find a combination of several autoimmune diseases in a single patient, given their common background. In our case, MPA was diagnosed in a patient with a prior history of primary biliary cirrhosis, even though there are few references in the literature of cases in which these two diseases are combined.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">5–7</span></a></p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Ruiz-Ferreras E, Martín-Arribas A, Tabernero-Fernández G, Lerma-Márquez JL, Fraile-Gómez P, García-Cosmes P, et al. Poliangeítis microscópica en paciente con cirrosis biliar primaria. Complicaciones del tratamiento. Nefrología. 2016;36:78–80.</p>" ] ] "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" => 1398 "Ancho" => 2248 "Tamanyo" => 128843 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Evolution of creatinine, proteinuria and p-ANCA figures after starting treatment with corticosteroids and cyclophosphamide. The grey band represents the period of admission in the nephrology department.</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" => 780 "Ancho" => 996 "Tamanyo" => 97065 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Colonoscopy.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:7 [ 0 => array:3 [ "identificador" => "bib0040" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "A case of granulomatosis with polyangiitis complicated by cyclophosphamide toxicity and opportunistic infections: choosing between Scylla and Charybdis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "E. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 9 | 6 | 15 |
| 2024 October | 41 | 48 | 89 |
| 2024 September | 37 | 26 | 63 |
| 2024 August | 49 | 47 | 96 |
| 2024 July | 32 | 26 | 58 |
| 2024 June | 49 | 35 | 84 |
| 2024 May | 58 | 33 | 91 |
| 2024 April | 50 | 32 | 82 |
| 2024 March | 31 | 27 | 58 |
| 2024 February | 41 | 38 | 79 |
| 2024 January | 31 | 27 | 58 |
| 2023 December | 22 | 26 | 48 |
| 2023 November | 32 | 31 | 63 |
| 2023 October | 31 | 36 | 67 |
| 2023 September | 22 | 23 | 45 |
| 2023 August | 35 | 31 | 66 |
| 2023 July | 40 | 22 | 62 |
| 2023 June | 35 | 20 | 55 |
| 2023 May | 33 | 43 | 76 |
| 2023 April | 17 | 17 | 34 |
| 2023 March | 34 | 23 | 57 |
| 2023 February | 26 | 13 | 39 |
| 2023 January | 30 | 27 | 57 |
| 2022 December | 52 | 34 | 86 |
| 2022 November | 42 | 36 | 78 |
| 2022 October | 48 | 39 | 87 |
| 2022 September | 53 | 33 | 86 |
| 2022 August | 53 | 41 | 94 |
| 2022 July | 33 | 40 | 73 |
| 2022 June | 35 | 34 | 69 |
| 2022 May | 48 | 29 | 77 |
| 2022 April | 64 | 52 | 116 |
| 2022 March | 38 | 49 | 87 |
| 2022 February | 67 | 40 | 107 |
| 2022 January | 59 | 31 | 90 |
| 2021 December | 32 | 39 | 71 |
| 2021 November | 39 | 38 | 77 |
| 2021 October | 81 | 42 | 123 |
| 2021 September | 35 | 26 | 61 |
| 2021 August | 44 | 34 | 78 |
| 2021 July | 50 | 34 | 84 |
| 2021 June | 67 | 21 | 88 |
| 2021 May | 52 | 45 | 97 |
| 2021 April | 90 | 24 | 114 |
| 2021 March | 70 | 30 | 100 |
| 2021 February | 45 | 36 | 81 |
| 2021 January | 39 | 20 | 59 |
| 2020 December | 44 | 26 | 70 |
| 2020 November | 42 | 22 | 64 |
| 2020 October | 35 | 23 | 58 |
| 2020 September | 29 | 11 | 40 |
| 2020 August | 39 | 7 | 46 |
| 2020 July | 37 | 14 | 51 |
| 2020 June | 38 | 10 | 48 |
| 2020 May | 36 | 9 | 45 |
| 2020 April | 37 | 27 | 64 |
| 2020 March | 20 | 10 | 30 |
| 2020 February | 37 | 25 | 62 |
| 2020 January | 41 | 17 | 58 |
| 2019 December | 42 | 29 | 71 |
| 2019 November | 38 | 12 | 50 |
| 2019 October | 17 | 12 | 29 |
| 2019 September | 29 | 12 | 41 |
| 2019 August | 22 | 17 | 39 |
| 2019 July | 22 | 23 | 45 |
| 2019 June | 32 | 11 | 43 |
| 2019 May | 37 | 26 | 63 |
| 2019 April | 74 | 29 | 103 |
| 2019 March | 39 | 25 | 64 |
| 2019 February | 23 | 18 | 41 |
| 2019 January | 37 | 20 | 57 |
| 2018 December | 129 | 38 | 167 |
| 2018 November | 178 | 18 | 196 |
| 2018 October | 171 | 32 | 203 |
| 2018 September | 94 | 17 | 111 |
| 2018 August | 68 | 17 | 85 |
| 2018 July | 84 | 12 | 96 |
| 2018 June | 96 | 20 | 116 |
| 2018 May | 87 | 9 | 96 |
| 2018 April | 136 | 15 | 151 |
| 2018 March | 119 | 7 | 126 |
| 2018 February | 104 | 7 | 111 |
| 2018 January | 101 | 5 | 106 |
| 2017 December | 96 | 6 | 102 |
| 2017 November | 77 | 18 | 95 |
| 2017 October | 42 | 8 | 50 |
| 2017 September | 35 | 11 | 46 |
| 2017 August | 26 | 11 | 37 |
| 2017 July | 36 | 10 | 46 |
| 2017 June | 41 | 17 | 58 |
| 2017 May | 35 | 13 | 48 |
| 2017 April | 40 | 10 | 50 |
| 2017 March | 25 | 5 | 30 |
| 2017 February | 44 | 7 | 51 |
| 2017 January | 13 | 9 | 22 |
| 2016 December | 29 | 7 | 36 |
| 2016 November | 59 | 13 | 72 |
| 2016 October | 69 | 3 | 72 |
| 2016 September | 94 | 2 | 96 |
| 2016 August | 70 | 2 | 72 |
| 2016 July | 149 | 11 | 160 |
| 2016 June | 110 | 0 | 110 |
| 2016 May | 122 | 0 | 122 |
| 2016 April | 72 | 0 | 72 |
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