Case Letter
A case of calciphylaxis with an unfavorable outcome
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0 ] "pt" => array:10 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Carta – Caso clínico</span>" "titulo" => "Um caso de calcifilaxia com desfecho desfavorável" "tienePdf" => "pt" "tieneTextoCompleto" => "pt" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "671" "paginaFinal" => "673" ] ] "contieneTextoCompleto" => array:1 [ "pt" => true ] "contienePdf" => array:1 [ "pt" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figura 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 546 "Ancho" => 905 "Tamanyo" => 64493 ] ] "descripcion" => array:1 [ "pt" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Placas eritematovioláceas, irregulares, com centro necrótico, localizadas no terço distal dos membros inferiores.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ 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array:1 [ "en" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1040 "Ancho" => 905 "Tamanyo" => 99349 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Physical examination revealed bilateral, strikingly symmetric, well-delimited eroded plaques on both breasts.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Fernando Garcia-Souto" "autores" => array:1 [ 0 => array:2 [ "nombre" => "Fernando" "apellidos" => "Garcia-Souto" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "pt" => array:9 [ "pii" => "S2666275220302587" "doi" => "10.1016/j.abdp.2020.03.006" "estado" 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"autores" => array:4 [ 0 => array:4 [ "nombre" => "Maria Carolina Ribeiro" "apellidos" => "Braga" "email" => array:1 [ 0 => "mariacarolbraga@hotmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "Susana" "apellidos" => "Strougo" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "Enoï Guedes" "apellidos" => "Vilar" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "Sandra Maria Barbosa" "apellidos" => "Durães" "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Department of Dermatology, Universidade Federal Fluminense, Niterói, RJ, Brazil" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Department of Dermatopathology, Universidade Federal Fluminense, Niterói, RJ, Brazil" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 672 "Ancho" => 905 "Tamanyo" => 145607 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Positive Von Kossa stain on the material deposited in the dermis and blood vessel wall (arrows; ×100).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Calciphylaxis or calcifying uremic arteriolopathy is a rare and serious complication secondary to late-stage chronic kidney disease (CKD).<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> It is observed in 1% to 4.5% of dialysis patients, especially those undergoing hemodialysis and women.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Hyperparathyroidism (HPT) secondary to nephropathy leads to changes in the metabolism of calcium (Ca) and phosphorus (P), representing the main etiological factor.</p><p id="par0010" class="elsevierStylePara elsevierViewall">The authors report the case of a female patient, aged 50 years, with a history of painful skin lesion with ten days of evolution (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>), soon followed by a necrotic ulcer in the lower limbs. The patient had CKD secondary to diabetes mellitus and arterial hypertension and had undergone peritoneal dialysis for five years. Dermatological examination showed irregular, erythematous-violaceous plaques, with central necrotic ulcers, measuring from 5 to 10 cm, in the distal third of the lower limbs (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Laboratory investigation revealed an increase in P, Ca, parathyroid hormone (PTH), and alkaline phosphatase, as well as anemia and hypoalbuminemia. Histological examination of the edge of an ulcer indicated a slight superficial perivascular inflammatory infiltrate and foci of Ca deposition in the subcutaneous tissue and vascular wall, confirmed by Von Kossa staining (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). Leg radiography revealed vascular calcifications in the topography of the popliteal and posterior tibial arteries, in addition to a slight diffuse increase in soft tissue density. The diagnosis of calciphylaxis was made, based on clinical, radiological, and histopathological findings. Treatment was started with a hypophosphatemic diet and adjustments in the dialysis to correct Ca, P, and PTH, analgesia, and dressing with 1% chloramphenicol on the ulcers. After failure of clinical treatment, surgical debridement and antibiotic therapy with amoxicillin and potassium clavulanate were performed. Due to poor response, the antibiotic drugs were replaced with vancomycin hydrochloride. Despite this conduct, an increase in the necrotic area was observed, and the patient’s clinical condition deteriorated, leading to death due to septic shock.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Calciphylaxis is associated with changes in the metabolism of Ca and P due to HPT secondary to CKD.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> In this context, deficits in calcitriol and abnormalities of Ca and vitamin D receptors favor PTH hypersecretion and disease progression. Risk factors include obesity, diabetes, liver disease, use of systemic corticosteroids, and a result of Ca × P above 70 mg/dL.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Very few cases have been described in patients without renal dysfunction; malignant neoplasia, severe liver disease, inflammatory bowel disease, and primary HPT should be ruled out.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">Its etiopathogenesis is not fully established and there is calcification and obstruction of the small and medium caliber blood vessels of the dermis, intima and media hyperplasia, and septal and/or lobular subcutaneous necrosis leading to distal ischemia and necrotic ulcers.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Systemic ischemic impairment, such as acute myocardial infarction, stroke, mesenteric ischemia, and peripheral arterial obstructions, may also occur.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Skin lesions are described as painful erythematous-violaceous macules similar to livedo reticularis, progressing to erythematous subcutaneous plaques or nodules with violaceus periphery, which may evolve to central necrosis and ulceration, with no tendency to spontaneous healing and a predilection for the lower limbs.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Sites such as the breasts, buttocks, and abdomen can be affected, with a worse prognosis.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">The diagnosis is made on a clinical and laboratory basis, and can be confirmed by histopathology. Laboratory findings include increased PTH, Ca, P, alkaline phosphatase, creatinine, Ca × P product, and anemia. Vasculitis, systemic lupus erythematosus, antiphospholipid antibody syndrome, and Henoch-Schönlein purpura should be ruled out.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Normalization of serum Ca, P, and PTH levels is recommended through a hypophosphatemic diet, Ca-free P chelators, calcimimetics, and sodium thiosulfate.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Pain should be controlled with strict analgesia and systemic antibiotic therapy if necessary.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Parathyroidectomy is reserved for severe and refractory HPT.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> The local treatment of ulcers is based on occlusive dressings with fibrinolytics and antibiotics, surgical debridement, and even the use of a hyperbaric chamber.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Mortality reaches 80% and sepsis is the main cause of death, as observed in the present case.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Therefore, it is important to emphasize the need for this entity to be known by dermatologists and nephrologists, since recognizing risk factors and employing preventive measures decreases its occurrence; if the disease is not avoided, early treatment is decisive for reducing mortality and a better prognosis.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Financial support</span><p id="par0050" class="elsevierStylePara elsevierViewall">None declared.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Authors’ contributions</span><p id="par0055" class="elsevierStylePara elsevierViewall">Maria Carolina Ribeiro Braga: Conception and planning of the study; elaboration and writing of the manuscript; obtaining, analyzing, and interpreting the data; intellectual participation in propaedeutic and/or therapeutic conduct of studied cases; critical review of the literature.</p><p id="par0060" class="elsevierStylePara elsevierViewall">Susana Strougo: Conception and planning of the study; elaboration and writing of the manuscript; critical review of the literature.</p><p id="par0065" class="elsevierStylePara elsevierViewall">Enoï Guedes Vilar: Effective participation in research orientation; intellectual participation in propaedeutic and/or therapeutic conduct of studied cases; critical review of the manuscript.</p><p id="par0070" class="elsevierStylePara elsevierViewall">Sandra Maria Barbosa Durães: Approval of the final version of the manuscript; effective participation in research orientation; intellectual participation in propaedeutic and/or therapeutic conduct of studied cases; critical review of the manuscript.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of interest</span><p id="par0075" class="elsevierStylePara elsevierViewall">None declared.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Financial support" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Authors’ contributions" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Conflicts of interest" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2019-11-28" "fechaAceptado" => "2020-02-27" "NotaPie" => array:2 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">How to cite this article: Braga MCR, Strougo S, Vilar EG, Durães SMB. A case of calciphylaxis with an unfavorable outcome. An Bras Dermatol. 2020; 95. <span class="elsevierStyleInterRef" id="intr0005" href="https://doi.org/10.1016/j.abd.2020.02.012">https://doi.org/10.1016/j.abd.2020.02.012</span>.</p>" ] 1 => array:2 [ "etiqueta" => "☆☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0010">Study conducted at the Dermatology Department, Hospital Universitário Antônio Pedro, Universidade Federal Fluminense, Niterói, RJ, Brazil.</p>" ] ] "multimedia" => array:3 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 546 "Ancho" => 905 "Tamanyo" => 63973 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Irregular erythematous-violaceous plaques, with necrotic centers, located in the distal third of the lower limbs.</p>" ] ] 1 => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1119 "Ancho" => 905 "Tamanyo" => 87483 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Evolution of the initial lesions to deep ulcerations with irregular and necrotic edges.</p>" ] ] 2 => array:8 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 672 "Ancho" => 905 "Tamanyo" => 145607 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Positive Von Kossa stain on the material deposited in the dermis and blood vessel wall (arrows; ×100).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Calciphylaxis: a rare but potentially fatal event of chronic kidney disease. 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| 2024 February | 94 | 71 | 165 |
| 2024 January | 73 | 47 | 120 |
| 2023 December | 66 | 60 | 126 |
| 2023 November | 71 | 74 | 145 |
| 2023 October | 70 | 67 | 137 |
| 2023 September | 75 | 77 | 152 |
| 2023 August | 60 | 33 | 93 |
| 2023 July | 66 | 38 | 104 |
| 2023 June | 68 | 42 | 110 |
| 2023 May | 42 | 24 | 66 |
| 2023 April | 30 | 24 | 54 |
| 2023 March | 53 | 34 | 87 |
| 2023 February | 35 | 38 | 73 |
| 2023 January | 34 | 26 | 60 |
| 2022 December | 42 | 26 | 68 |
| 2022 November | 63 | 47 | 110 |
| 2022 October | 93 | 42 | 135 |
| 2022 September | 49 | 51 | 100 |
| 2022 August | 37 | 56 | 93 |
| 2022 July | 36 | 51 | 87 |
| 2022 June | 40 | 53 | 93 |
| 2022 May | 26 | 46 | 72 |
| 2022 April | 30 | 55 | 85 |
| 2022 March | 43 | 56 | 99 |
| 2022 February | 32 | 25 | 57 |
| 2022 January | 70 | 71 | 141 |
| 2021 December | 31 | 48 | 79 |
| 2021 November | 31 | 51 | 82 |
| 2021 October | 56 | 64 | 120 |
| 2021 September | 26 | 37 | 63 |
| 2021 August | 39 | 50 | 89 |
| 2021 July | 27 | 27 | 54 |
| 2021 June | 33 | 49 | 82 |
| 2021 May | 48 | 92 | 140 |
| 2021 April | 91 | 199 | 290 |
| 2021 March | 116 | 49 | 165 |
| 2021 February | 51 | 17 | 68 |
| 2021 January | 38 | 22 | 60 |
| 2020 December | 41 | 18 | 59 |
| 2020 November | 20 | 17 | 37 |
| 2020 October | 26 | 9 | 35 |
| 2020 September | 19 | 11 | 30 |
| 2020 August | 14 | 6 | 20 |
| 2020 July | 4 | 5 | 9 |
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