Images in Clinical Rheumatology
Ischiopubic Pain in a Patient with Paget's Disease
Dolor en la región isquiopubiana en un paciente con enfermedad de Paget
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Suárez, Gloria Alonso Claudio, Leticia Moralejo Alonso, Antonio Jiménez López" "autores" => array:6 [ 0 => array:4 [ "nombre" => "Ignacio" "apellidos" => "Novo Veleiro" "email" => array:1 [ 0 => "ignacio.novo.veleiro@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" => "María Electra" "apellidos" => "Perianes Díaz" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "María Lucía" "apellidos" => "Alvela Suárez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "Gloria" "apellidos" => "Alonso Claudio" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => 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"etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Dolor en la región isquiopubiana en un paciente con enfermedad de Paget" ] ] "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" => 844 "Ancho" => 900 "Tamanyo" => 117696 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Multiple lesions compatible with Paget's disease and a larger lytic image on the left ischium.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Clinical Case</span><p id="par0005" class="elsevierStylePara elsevierViewall">We report the case of an 82-year-old male patient diagnosed 10 years prior with Paget's disease and polyostotic involvement. Relevant history showed atrial fibrillation and chronic heart failure. He came to the immediate care clinic (UCAI) due to fatigue, anorexia and edema of the lower extremities, which had been present for 15 days. The systems review referred pain in the left ischiopubic region since 6 months before, which had been increasing and appeared both at rest and with exercise. He was admitted for heart failure treatment and study of the origin of pain. Physical examination showed pale skin and mucous membranes, irregular cardiac auscultation with systolic aortic murmur, decreased breath sounds on widespread lung auscultation, pitting edema up to the root of both lower limbs and pain on movement and decreased function of the left hip joint. On rectal examination there were no masses but only with remnants of normal stool. Complementary tests showed hemoglobin of 9.5<span class="elsevierStyleHsp" style=""></span>g/dl with normal mean corpuscular volume and hemoglobin, CRP: 21.2<span class="elsevierStyleHsp" style=""></span>mg/dl (0.2–0.8), fibrinogen: 722<span class="elsevierStyleHsp" style=""></span>mg/dl (200–400), glomerular filtration rate: 120<span class="elsevierStyleHsp" style=""></span>mm (1–15), alkaline phosphatase, 155<span class="elsevierStyleHsp" style=""></span>U/l, iron: 23.6<span class="elsevierStyleHsp" style=""></span>mg/dl (50–150) transferrin: 155<span class="elsevierStyleHsp" style=""></span>mg/dl (300–360), ferritin: 1144<span class="elsevierStyleHsp" style=""></span>mg/l (50–350), saturation index: 12% (20–40), soluble transferrin receptor: 7.88<span class="elsevierStyleHsp" style=""></span>mg/l (2–4), tumor markers were normal. The chest X-ray showed bilateral pleural effusion and small lesions consistent with Paget's disease in both shoulders and the fifth left rib. Pelvic X-ray showed multiple lesions consistent with Paget's disease and a larger lytic image on the left ischium (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). A bone scan was performed which showed images compatible with Paget's disease and no uptake in the left ischium (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). A thoraco-abdominal CT-scan revealed a large pelvic mass with lobulated contours and foci of calcification that destroyed the left ischium and acetabulum measuring 12<span class="elsevierStyleHsp" style=""></span>cm×12<span class="elsevierStyleHsp" style=""></span>cm×16<span class="elsevierStyleHsp" style=""></span>cm (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). Suspecting a bone neoplasm added to Paget's disease the patient underwent an iliac bone biopsy which was consistent with undifferentiated pleomorphic high grade non osteogenic sarcoma, vimentin positive, with coexpression of CD-68 and low expression of actin and desmin. The patient outcome was unfavorable, suffering great malaise and difficulty controlling the pain, he decided to initiate treatment with radiotherapy for symptomatic control fundamentally. Finally, the patient died a week after the pathological diagnosis.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Discussion</span><p id="par0010" class="elsevierStylePara elsevierViewall">Paget's disease, described in 1876 by Sir James Paget, is characterized by excessive and abnormal bone remodeling, altering both the resorption and bone formation and leading to the appearance of lytic lesions associated with hypetrophic bone deformities.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Although the pathogenesis of the disease remains unknown, it has been found associated with the expression of certain genes,<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a> some of which, like SQSTM1, could also be related to the development of osteosarcomas on prior lesions.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,4</span></a> This neoplasia may develop in up to 1% of cases according to different series.<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,5</span></a> Other, more sporadic tumors of different cell lines may be found in these patients, such as bone malignant fibrous histiocytoma or undifferentiated pleomorphic sarcoma as in our case.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> This tumor is rare, constituting 2%–5% of malignant bone tumors, and usually appears as a palpable and painful mass on the femur, tibia or humerus, with large lytic lesions on X-rays. This is the clinical presentation described in previously reported cases, similar to the one presented, but usually the alkaline phosphatase elevation is much higher than in our case.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> Its most common presentation as the primary tumor, but in 20%–30% of cases it appears in connection with pre-existing bone lesions, such as those caused by Paget's disease, radiotherapy treatments, joint replacement, trauma, stroke or bone infections.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">7–11</span></a> Treatment is surgical and adjuvant radiotherapy may be used, though recent studies show that the response to chemotherapy may be similar to that of osteosarcoma, aggressive treatment usually points to a poor prognosis and high mortality, partly due to its high rate of recurrence (around 65%).<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a></p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Disclosures</span><p id="par0015" class="elsevierStylePara elsevierViewall">The authors have no disclosures to make.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Clinical Case" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Discussion" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Disclosures" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara">Please cite this article as: Novo Veleiro I, et al. Dolor en la región isquiopubiana en un paciente con enfermedad de Paget. Reumatol Clin. 2011;<span class="elsevierStyleBold">7(6)</span>:419–20.</p>" ] ] "multimedia" => array:3 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 844 "Ancho" => 900 "Tamanyo" => 117696 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Multiple lesions compatible with Paget's disease and a larger lytic image on the left ischium.</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" => 304 "Ancho" => 400 "Tamanyo" => 56625 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Bone scan showing images compatible with Paget's disease and the absence of uptake on the left ischium.</p>" ] ] 2 => array:7 [ "identificador" => "fig0015" "etiqueta" => "Fig. 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 843 "Ancho" => 900 "Tamanyo" => 87897 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Thoraco-abdominal-pelvic CT showing a great mass with lobulated contour and foci of calcification destroying the ischium and left acetabulum, measuring 12<span class="elsevierStyleHsp" style=""></span>cm×12<span class="elsevierStyleHsp" style=""></span>cm×16<span class="elsevierStyleHsp" style=""></span>cm.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:11 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Paget's disease of bone: a review" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "M. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 12 | 10 | 22 |
| 2024 October | 54 | 27 | 81 |
| 2024 September | 60 | 14 | 74 |
| 2024 August | 62 | 26 | 88 |
| 2024 July | 63 | 26 | 89 |
| 2024 June | 58 | 43 | 101 |
| 2024 May | 64 | 29 | 93 |
| 2024 April | 57 | 16 | 73 |
| 2024 March | 69 | 27 | 96 |
| 2024 February | 43 | 21 | 64 |
| 2024 January | 52 | 19 | 71 |
| 2023 December | 50 | 23 | 73 |
| 2023 November | 77 | 24 | 101 |
| 2023 October | 51 | 28 | 79 |
| 2023 September | 81 | 31 | 112 |
| 2023 August | 52 | 19 | 71 |
| 2023 July | 25 | 19 | 44 |
| 2023 June | 80 | 24 | 104 |
| 2023 May | 62 | 17 | 79 |
| 2023 April | 45 | 8 | 53 |
| 2023 March | 84 | 33 | 117 |
| 2023 February | 58 | 28 | 86 |
| 2023 January | 40 | 17 | 57 |
| 2022 December | 95 | 40 | 135 |
| 2022 November | 73 | 30 | 103 |
| 2022 October | 61 | 39 | 100 |
| 2022 September | 74 | 29 | 103 |
| 2022 August | 55 | 36 | 91 |
| 2022 July | 64 | 36 | 100 |
| 2022 June | 67 | 37 | 104 |
| 2022 May | 87 | 45 | 132 |
| 2022 April | 73 | 52 | 125 |
| 2022 March | 72 | 42 | 114 |
| 2022 February | 118 | 33 | 151 |
| 2022 January | 60 | 30 | 90 |
| 2021 December | 41 | 33 | 74 |
| 2021 November | 65 | 50 | 115 |
| 2021 October | 61 | 56 | 117 |
| 2021 September | 79 | 48 | 127 |
| 2021 August | 68 | 55 | 123 |
| 2021 July | 53 | 35 | 88 |
| 2021 June | 77 | 38 | 115 |
| 2021 May | 56 | 41 | 97 |
| 2021 April | 129 | 82 | 211 |
| 2021 March | 101 | 31 | 132 |
| 2021 February | 45 | 19 | 64 |
| 2021 January | 42 | 23 | 65 |
| 2020 December | 50 | 13 | 63 |
| 2020 November | 28 | 25 | 53 |
| 2020 October | 29 | 15 | 44 |
| 2020 September | 38 | 21 | 59 |
| 2020 August | 29 | 13 | 42 |
| 2020 July | 19 | 11 | 30 |
| 2020 June | 32 | 20 | 52 |
| 2020 May | 39 | 21 | 60 |
| 2020 April | 18 | 19 | 37 |
| 2020 March | 25 | 7 | 32 |
| 2020 January | 4 | 0 | 4 |
| 2019 September | 7 | 0 | 7 |
| 2019 June | 2 | 0 | 2 |
| 2019 March | 2 | 0 | 2 |
| 2019 January | 1 | 0 | 1 |
| 2018 May | 6 | 0 | 6 |
| 2018 April | 71 | 5 | 76 |
| 2018 March | 75 | 4 | 79 |
| 2018 February | 54 | 4 | 58 |
| 2018 January | 49 | 4 | 53 |
| 2017 December | 62 | 4 | 66 |
| 2017 November | 50 | 5 | 55 |
| 2017 October | 52 | 5 | 57 |
| 2017 September | 55 | 5 | 60 |
| 2017 August | 51 | 4 | 55 |
| 2017 July | 57 | 8 | 65 |
| 2017 June | 87 | 13 | 100 |
| 2017 May | 99 | 5 | 104 |
| 2017 April | 64 | 5 | 69 |
| 2017 March | 68 | 3 | 71 |
| 2017 February | 117 | 8 | 125 |
| 2017 January | 55 | 5 | 60 |
| 2016 December | 85 | 10 | 95 |
| 2016 November | 78 | 5 | 83 |
| 2016 October | 111 | 12 | 123 |
| 2016 September | 185 | 4 | 189 |
| 2016 August | 106 | 9 | 115 |
| 2016 July | 37 | 7 | 44 |
| 2016 June | 0 | 8 | 8 |
| 2016 May | 0 | 6 | 6 |
| 2016 April | 0 | 4 | 4 |
| 2016 February | 4 | 0 | 4 |
| 2016 January | 1 | 0 | 1 |
| 2015 December | 3 | 11 | 14 |
| 2015 November | 1 | 0 | 1 |
| 2015 October | 1 | 11 | 12 |
| 2015 September | 4 | 0 | 4 |
| 2015 August | 12 | 0 | 12 |
| 2015 July | 48 | 4 | 52 |
| 2015 June | 45 | 7 | 52 |
| 2015 May | 61 | 18 | 79 |
| 2015 April | 41 | 12 | 53 |
| 2015 March | 65 | 8 | 73 |
| 2015 February | 61 | 8 | 69 |
| 2015 January | 58 | 9 | 67 |
| 2014 December | 66 | 8 | 74 |
| 2014 November | 58 | 6 | 64 |
| 2014 October | 54 | 10 | 64 |
| 2014 September | 56 | 9 | 65 |
| 2014 August | 68 | 18 | 86 |
| 2014 July | 50 | 18 | 68 |
| 2014 June | 58 | 9 | 67 |
| 2014 May | 69 | 19 | 88 |
| 2014 April | 66 | 8 | 74 |
| 2014 March | 84 | 13 | 97 |
| 2014 February | 64 | 11 | 75 |
| 2014 January | 68 | 10 | 78 |
| 2013 December | 64 | 10 | 74 |
| 2013 November | 61 | 11 | 72 |
| 2013 October | 73 | 6 | 79 |
| 2013 September | 57 | 13 | 70 |
| 2013 August | 65 | 12 | 77 |
| 2013 July | 50 | 9 | 59 |
| 2013 June | 53 | 12 | 65 |
| 2013 May | 52 | 15 | 67 |
| 2013 April | 51 | 15 | 66 |
| 2013 March | 64 | 15 | 79 |
| 2013 February | 35 | 8 | 43 |
| 2013 January | 41 | 9 | 50 |
| 2012 December | 34 | 6 | 40 |
| 2012 November | 32 | 13 | 45 |
| 2012 October | 41 | 5 | 46 |
| 2012 September | 19 | 2 | 21 |
| 2012 July | 4 | 0 | 4 |
| 2012 June | 34 | 0 | 34 |
| 2012 May | 20 | 0 | 20 |
| 2012 April | 22 | 0 | 22 |
| 2012 March | 43 | 0 | 43 |
| 2012 February | 31 | 0 | 31 |
| 2012 January | 3 | 0 | 3 |
| 2011 December | 4 | 0 | 4 |
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