Letter to the Editor
Collagen type III nephropathy as a systemic disease? – A case report
Glomerulopatía por colágeno tipo III como enfermedad sistémica: caso clínico
a University Medical Centre Maribor, Division of Internal Medicine, Department of Nephrology, Ljubljanska 5, Maribor, Slovenia
b University Medical Centre Maribor, Division of Internal Medicine, Department of Dialysis, Ljubljanska 5, Maribor, Slovenia
c University of Maribor, Faculty of Medicine, Taborska 5, Maribor, Slovenia
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A) Trombos en los capilares glomerulares y arterio aferente, necrosis fibrinoide, colapso de los capilares glomerulares (H&E). B) Trombos en los capilares glomerulares y arteriolas (Masson). C) Tinción CD61<span class="elsevierStyleSup">+</span> en los capilares glomerulares. D) Tinción C4d<span class="elsevierStyleSup">+</span> en los capilares peritubulares.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Teresa Bada-Bosch, Beatriz Redondo, Angel M. Sevillano, Marina Alonso, Hernando Trujillo, Pilar Auñón, Natalia Polanco, Lucía Rodríguez, Esther Gonzalez, Amado Andrés" "autores" => array:10 [ 0 => array:2 [ "nombre" => "Teresa" "apellidos" => "Bada-Bosch" ] 1 => array:2 [ "nombre" => "Beatriz" "apellidos" => "Redondo" ] 2 => array:2 [ "nombre" => "Angel M." 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(a, b) Violaceous, plaque-like subcutaneous nodules surrounded by livedo reticularis-like patches and necrotic ulcerations on legs. (c) Sclerotic subcutaneous nodules and peau d’orange appearance on infraumblical region.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Muge Gore Karaali, Vefa Aslı Turgut Erdemir, Cem Leblebici, Ozan Erdem, Asude Kara Polat, Mehmet Salih Gurel" "autores" => array:6 [ 0 => array:2 [ "nombre" => "Muge" "apellidos" => "Gore Karaali" ] 1 => array:2 [ "nombre" => "Vefa Aslı" "apellidos" => "Turgut Erdemir" ] 2 => array:2 [ "nombre" => "Cem" "apellidos" => "Leblebici" ] 3 => array:2 [ "nombre" => "Ozan" "apellidos" => "Erdem" ] 4 => array:2 [ "nombre" => "Asude" "apellidos" => "Kara Polat" ] 5 => array:2 [ "nombre" => "Mehmet Salih" "apellidos" => "Gurel" ] ] ] ] ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0211699519300931?idApp=UINPBA000064" "url" => "/02116995/0000004000000001/v1_202001221359/S0211699519300931/v1_202001221359/en/main.assets" ] "en" => array:14 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Letter to the Editor</span>" "titulo" => "Collagen type III nephropathy as a systemic disease? – A case report" "tieneTextoCompleto" => true "saludo" => "Dear Editor:" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "106" "paginaFinal" => "108" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Eva Jakopin, Sebastjan Bevc, Robert Ekart, Radovan Hojs" "autores" => array:4 [ 0 => array:4 [ "nombre" => "Eva" "apellidos" => "Jakopin" "email" => array:1 [ 0 => "eva.jakopin@ukc-mb.si" ] "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" => "Sebastjan" "apellidos" => "Bevc" "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] 2 => array:3 [ "nombre" => "Robert" "apellidos" => "Ekart" "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] 3 => array:3 [ "nombre" => "Radovan" "apellidos" => "Hojs" "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "University Medical Centre Maribor, Division of Internal Medicine, Department of Nephrology, Ljubljanska 5, Maribor, Slovenia" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "University Medical Centre Maribor, Division of Internal Medicine, Department of Dialysis, Ljubljanska 5, Maribor, Slovenia" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "University of Maribor, Faculty of Medicine, Taborska 5, Maribor, Slovenia" "etiqueta" => "c" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Glomerulopatía por colágeno tipo III como enfermedad sistémica: caso clínico" ] ] "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" => 680 "Ancho" => 905 "Tamanyo" => 140107 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Brown staining of collagen III in the interstitial space, in expanded mesangium and in subendothelial areas (imunoperoxidase reaction with anti-collagen III antibody, 60×).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">We present a case of a 57-year old female who complained of progressive weight loss, general weakness, dysphagia and mild bilateral leg swelling. She had a history of asthma, arterial hypertension, hypothyrosis due to Hashimoto thyroiditis and axonal sensorimotor polyneuropathy of unknown cause. She had no family history of renal disease. General and systemic examination was unremarkable except minimal peripheral leg edema and mildly atrophic lower limb musculature. She had no nail or patella dysplasia.</p><p id="par0010" class="elsevierStylePara elsevierViewall">Blood test results: creatinine 110<span class="elsevierStyleHsp" style=""></span>μmol/L; CKD-EPI glomerular filtration rate 48<span class="elsevierStyleHsp" style=""></span>ml/min/1.73<span class="elsevierStyleHsp" style=""></span>m<span class="elsevierStyleSup">2</span>, cystatin C 2.8<span class="elsevierStyleHsp" style=""></span>mg/dL, mildly elevated gamma glutamyl transferase and alkaline phosphatase, hypoalbuminemia 32<span class="elsevierStyleHsp" style=""></span>g/L, positive ANA H+ (1:160).</p><p id="par0015" class="elsevierStylePara elsevierViewall">Urine test results: microhematuria and proteinuria 4<span class="elsevierStyleHsp" style=""></span>g per day.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Diagnostic procedures revealed mild to moderate pulmonary hypertension, mild pericardial effusion, hepatosplenomegaly, bilateral minimal pleural effusion and ascites (exudate with negative cytology results).</p><p id="par0025" class="elsevierStylePara elsevierViewall">Renal biopsy revealed a diffuse endoproliferative (mesangioproliferative pattern) exudative sclerosing glomerulonephritis with collagen III deposition within mesangium and capillary walls, accompanied with diffuse chronic fibrosing mononuclear tubulo-interstitial nephritis. Immunocomplex glomerulonephritises were excluded (<a class="elsevierStyleCrossRefs" href="#fig0005">Figs. 1 and 2</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">Measurement of N-terminal peptide of type III procollagen (PIIIP) in serum (19.2<span class="elsevierStyleHsp" style=""></span>ng/mL) and urine (53.6<span class="elsevierStyleHsp" style=""></span>ng/mL) was performed (RIA-gnost® PIIIP kit, Cisbio Bioassays).</p><p id="par0035" class="elsevierStylePara elsevierViewall">Our patient was discharged with furosemide and angiotensin-converting-enzyme (ACE) inhibitor. On follow-up her renal function was stable. In a few months she died at home. Unfortunately the autopsy was not performed and the cause of death remained unknown.</p><p id="par0040" class="elsevierStylePara elsevierViewall">Collagen type III nephropathy (Col3NP) is a rare glomerular disease with characteristic accumulation of abnormal collagen type III in glomerular mesangium and subendothelial space and increase in serum PIIIP levels.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">1</span></a> The etiopathogenesis remains unclear, but ethnic and/or genetic factors may play a role.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">2</span></a> Some authors suggested autosomal recessive inheritance pattern, whereas in our case the occurrence of disease was sporadic.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">3</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The most common clinical presentation is proteinuria, which is in the nephrotic range in more than 50% of the patients, with or without associated nephrotic syndrome. Hematuria is microscopic. Arterial hypertension is present in approximately 60% of the patients. Renal function is usually normal or slightly reduced at presentation, but in 50% there is progression to end-stage renal disease (ESRD).<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">3</span></a> Our patient presented with nephrotic range proteinuria and microscopic hematuria but without arterial hypertension. She had chronic kidney disease (CKD) stage 3. Our histology findings from renal biopsy were consistent with data from literature presenting patients with Col3NP.<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">2,3</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Type III collagen is a structural protein of extracellular matrix and is present in tissues showing elastic properties. In kidney it is normally present only in the interstitium and blood vessels, but not in the glomerulus.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">4</span></a> Although the pathogenesis of this disease is still unknown, two explanations have been proposed. One is the mesangial production of abnormal collagen, another is mesangial accumulation of type III collagen derived from other organs.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">5</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">The disease is mainly limited to kidneys, but there are a few isolated reports of extra-renal involvement with Col3NP leading to a hypothesis that renal lesion may be a part of a systemic disease with abnormal metabolism of type III collagen.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">5</span></a> Abnormal collagen III fibers have been found in liver, spleen and kidneys and in some other organs (myocardium, pancreas, thyroid gland, alveoli).<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">5</span></a> According to clinical examination and all pathological findings we believed that our patient had some kind of systemic disease. She had multiple organ damage which we could not explained otherwise. She developed hepatosplenomegaly with ascites. Without biopsy of the liver etiology of liver disease in our patient remained unknown, however we excluded other common causes (viral hepatitides, alcohol consumption, cirrhosis, hemochromatosis, cancer, intoxication, etc.). Since there is data from literature that collagen III deposition was found in spleen and liver in some patients with confirmed Col3NP, we assumed that was the cause in our patient as well. Furthermore, unexplained polyneuropathy of our patient may as well be caused by abnormal collagen III deposition in nervous system, since common neurological diseases were excluded. Additionally, there was also unexplained pulmonary hypertension.</p><p id="par0060" class="elsevierStylePara elsevierViewall">Unfortunately, Col3NP is a progressive disease and no specific therapy is available. Supportive measures include control of arterial hypertension, diuretics to relieve edema and renal replacement therapy if needed.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">4</span></a> Steroids, cyclosporine, cyclophosphamide and low-density lipoprotein apheresis had been tried with no benefit.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">6</span></a> The progression of the disease is assumed to depend on the extent of deposition and the amount of tubulointerstitial fibrosis and atrophy.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">7</span></a> In an isolated renal pathology the worst end point is progression to ESRD, whereas in a systemic involvement there is progression to multiorgan failure and death. We can speculate, that in a systemic type of disease with multiple organs affected, the progression is even faster. We assumed that was the case in our patient – she died a few months after the diagnosis.</p></span>" "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" => 680 "Ancho" => 905 "Tamanyo" => 223313 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Diffuse global proliferative glomerulonephritis with lobulation of glomeruli (HE, 20×).</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" => 680 "Ancho" => 905 "Tamanyo" => 140107 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Brown staining of collagen III in the interstitial space, in expanded mesangium and in subendothelial areas (imunoperoxidase reaction with anti-collagen III antibody, 60×).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:7 [ 0 => array:3 [ "identificador" => "bib0040" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Collagenofibrotic glomerulopathy – case report with review of literature" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "K.C. 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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 8 | 12 | 20 |
| 2024 Octubre | 52 | 43 | 95 |
| 2024 Septiembre | 39 | 26 | 65 |
| 2024 Agosto | 61 | 64 | 125 |
| 2024 Julio | 38 | 27 | 65 |
| 2024 Junio | 59 | 38 | 97 |
| 2024 Mayo | 55 | 32 | 87 |
| 2024 Abril | 63 | 40 | 103 |
| 2024 Marzo | 57 | 23 | 80 |
| 2024 Febrero | 34 | 29 | 63 |
| 2024 Enero | 49 | 23 | 72 |
| 2023 Diciembre | 46 | 32 | 78 |
| 2023 Noviembre | 49 | 38 | 87 |
| 2023 Octubre | 55 | 33 | 88 |
| 2023 Septiembre | 69 | 32 | 101 |
| 2023 Agosto | 54 | 29 | 83 |
| 2023 Julio | 46 | 17 | 63 |
| 2023 Junio | 39 | 21 | 60 |
| 2023 Mayo | 58 | 30 | 88 |
| 2023 Abril | 42 | 26 | 68 |
| 2023 Marzo | 62 | 23 | 85 |
| 2023 Febrero | 56 | 15 | 71 |
| 2023 Enero | 70 | 26 | 96 |
| 2022 Diciembre | 62 | 35 | 97 |
| 2022 Noviembre | 57 | 33 | 90 |
| 2022 Octubre | 64 | 44 | 108 |
| 2022 Septiembre | 64 | 27 | 91 |
| 2022 Agosto | 61 | 53 | 114 |
| 2022 Julio | 57 | 52 | 109 |
| 2022 Junio | 88 | 52 | 140 |
| 2022 Mayo | 91 | 43 | 134 |
| 2022 Abril | 100 | 63 | 163 |
| 2022 Marzo | 68 | 48 | 116 |
| 2022 Febrero | 72 | 45 | 117 |
| 2022 Enero | 42 | 36 | 78 |
| 2021 Diciembre | 79 | 52 | 131 |
| 2021 Noviembre | 66 | 38 | 104 |
| 2021 Octubre | 61 | 46 | 107 |
| 2021 Septiembre | 63 | 36 | 99 |
| 2021 Agosto | 112 | 50 | 162 |
| 2021 Julio | 68 | 41 | 109 |
| 2021 Junio | 89 | 35 | 124 |
| 2021 Mayo | 88 | 53 | 141 |
| 2021 Abril | 146 | 102 | 248 |
| 2021 Marzo | 106 | 56 | 162 |
| 2021 Febrero | 95 | 46 | 141 |
| 2021 Enero | 56 | 23 | 79 |
| 2020 Diciembre | 87 | 23 | 110 |
| 2020 Noviembre | 44 | 21 | 65 |
| 2020 Octubre | 43 | 14 | 57 |
| 2020 Septiembre | 58 | 29 | 87 |
| 2020 Agosto | 75 | 29 | 104 |
| 2020 Julio | 100 | 19 | 119 |
| 2020 Junio | 92 | 28 | 120 |
| 2020 Mayo | 74 | 33 | 107 |
| 2020 Abril | 52 | 28 | 80 |
| 2020 Marzo | 68 | 22 | 90 |
| 2020 Febrero | 130 | 40 | 170 |
| 2020 Enero | 68 | 24 | 92 |
| 2019 Diciembre | 24 | 18 | 42 |
| 2019 Noviembre | 14 | 10 | 24 |
| 2019 Octubre | 43 | 22 | 65 |
| 2019 Septiembre | 49 | 18 | 67 |
| 2019 Agosto | 62 | 42 | 104 |
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