Scientific Letter
Systemic Sclerosis and Kaposi's Sarcoma with Pulmonary Involvement: An Unexpected Association
Esclerosis sistémica y sarcoma de Kaposi con afectación pulmonar: una asociación inesperada
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(B) Histology of the pulmonary nodule sample, showing spindle cells characteristic of Kaposi's sarcoma.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Systemic sclerosis (SSc) is a rare disease, characterized by complex immune disorders, vascular damage and fibrosis of the skin and visceral organs.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> Autoimmune (AI) diseases, including SSc, and immunosuppressive drugs have been associated with many types of cancers.</p><p id="par0010" class="elsevierStylePara elsevierViewall">Kaposi's sarcoma (KS) is a rare angioproliferative tumor associated with human herpesvirus 8 (HHV-8) infection. Mucocutaneous involvement is the most common, but KS can be seen in almost all visceral sites.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a> Pulmonary KS can involve the lung parenchyma, airways, pleura, and/or intrathoracic lymph nodes.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> The iatrogenic type, one of the four KS types, is related to immunosuppressive therapy, typically in patients that have undergone solid organ transplantation or with other conditions such as AI diseases.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">We report the case of a 66-year-old woman, diagnosed with limited cutaneous SSc at the age of forty, with a four-year history of pulmonary involvement presenting as interstitial lung disease with an usual interstitial pneumonia pattern. She was being treated with pentoxifylline 1200<span class="elsevierStyleHsp" style=""></span>mg/day, pantoprazole 40<span class="elsevierStyleHsp" style=""></span>mg/day, azathioprine 100<span class="elsevierStyleHsp" style=""></span>mg/day (2<span class="elsevierStyleHsp" style=""></span>mg/kg/day) and prednisolone 10<span class="elsevierStyleHsp" style=""></span>mg/day.</p><p id="par0020" class="elsevierStylePara elsevierViewall">The patient presented a brownish-to-reddish <span class="elsevierStyleItalic">de novo</span> nodule on her lower right eyelid that was surgically removed, and which histological examination was compatible with KS. Serology for human immunodeficiency virus (HIV) was negative. Six months later, the patient presented some violaceous nodular lesions on her back, abdominal area and upper thigh, and reported worsening of respiratory symptoms, with more cough and nonpurulent sputum. She underwent high-resolution chest computed tomography that showed new multiple subcentimeter pulmonary nodules, diffusely dispersed throughout both lungs (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A). Routine laboratory tests showed no significant changes, and lymphocyte subpopulations on a peripheral blood sample were normal. Repeat HIV serology was again negative. Bronchofibroscopy with bronchoalveolar lavage was performed, revealing intense lymphocytosis, expected in the context of pulmonary involvement of SSc, but with negative microbiological examination and no signs of malignancy. A transthoracic needle biopsy of one of the pulmonary nodules was then carried out, and the histology showed characteristic spindle cells compatible with KS diagnosis (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>B). The latency-associated nuclear antigen (LANA-1) was not tested, but KS diagnosis was confirmed by HHV-8 DNA detected by polymerase chain reaction. Immunosuppressive therapy was adjusted to exclude azathioprine. The KS lesions remained stable and the patient was closely followed-up to detect progression of KS, which would have been treated with chemotherapy.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">To our knowledge, this is the first case of Kaposi's sarcoma with pulmonary involvement in a patient with SSc. Patients with SSc have been shown to have a higher prevalence of genetic damage that predisposes to carcinogenesis.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> Lung and hematological malignancies are more closely associated with SSc.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">KS has a very low incidence in patients with AI diseases.<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">4,5</span></a> However, it appears that the presence of an AI disease is in itself a risk factor for the development of KS, as some cases without prior immunosuppressants have been reported.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Furthermore, KS is known to be induced or triggered by immunosuppressive therapy in patients previously infected with HHV-8,<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">4,5</span></a> although no relationship between type, duration or required dosage has been established.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a> Studies have also shown that immunosuppression-related KS tends to be aggressive, with more frequent visceral involvement.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">In this case, the patient had been treated with a long-term combination of steroids with azathioprine, and had the particularity of presenting cutaneous and pulmonary KS. The extent to which pulmonary manifestations were associated with the presence of pulmonary involvement related to SSc is unknown, but there could be a link.</p><p id="par0045" class="elsevierStylePara elsevierViewall">In this context, the occurrence of KS in the absence of other predisposing factors, namely HIV infection, was interpreted to be treatment and/or disease related.</p><p id="par0050" class="elsevierStylePara elsevierViewall">With regard to the management of these patients, modification, reduction or withdrawal of the immunosuppressive medication should be attempted, as this will result in KS remission in about 50% of cases, while radiotherapy and/or chemotherapy may be needed in refractory or more severe cases.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</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: Aguiar F, Araújo D, Brito I. Esclerosis sistémica y sarcoma de Kaposi con afectación pulmonar: una asociación inesperada. Arch Bronconeumol. 2017;53:165–166.</p>" ] ] "multimedia" => array:1 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 539 "Ancho" => 1500 "Tamanyo" => 168410 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) High resolution chest computed tomography showing multiple bilateral nodules. (B) Histology of the pulmonary nodule sample, showing spindle cells characteristic of Kaposi's sarcoma.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0030" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Systemic sclerosis (scleroderma) and cancer risk: a systematic review and meta-analysis of observational studies" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "M. Bonifazi" 1 => "I. Tramacere" 2 => "G. Pomponio" 3 => "B. Gabrielli" 4 => "E.V. 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González" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.reuma.2012.07.002" "Revista" => array:6 [ "tituloSerie" => "Reumatol Clin" "fecha" => "2013" "volumen" => "9" "paginaInicial" => "243" "paginaFinal" => "245" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/23271139" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/15792129/0000005300000003/v1_201703240112/S1579212916301902/v1_201703240112/en/main.assets" "Apartado" => array:4 [ "identificador" => "49861" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Scientific letters" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/15792129/0000005300000003/v1_201703240112/S1579212916301902/v1_201703240112/en/main.pdf?idApp=UINPBA00003Z&text.app=https://archbronconeumol.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1579212916301902?idApp=UINPBA00003Z" ]
| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 5 | 1 | 6 |
| 2024 October | 73 | 17 | 90 |
| 2024 September | 64 | 24 | 88 |
| 2024 August | 67 | 46 | 113 |
| 2024 July | 63 | 23 | 86 |
| 2024 June | 63 | 33 | 96 |
| 2024 May | 90 | 27 | 117 |
| 2024 April | 56 | 29 | 85 |
| 2024 March | 60 | 16 | 76 |
| 2024 February | 45 | 20 | 65 |
| 2023 March | 14 | 3 | 17 |
| 2023 February | 63 | 15 | 78 |
| 2023 January | 41 | 26 | 67 |
| 2022 December | 73 | 36 | 109 |
| 2022 November | 83 | 28 | 111 |
| 2022 October | 74 | 39 | 113 |
| 2022 September | 52 | 28 | 80 |
| 2022 August | 54 | 43 | 97 |
| 2022 July | 49 | 36 | 85 |
| 2022 June | 43 | 38 | 81 |
| 2022 May | 60 | 36 | 96 |
| 2022 April | 56 | 29 | 85 |
| 2022 March | 63 | 26 | 89 |
| 2022 February | 63 | 38 | 101 |
| 2022 January | 88 | 37 | 125 |
| 2021 December | 92 | 50 | 142 |
| 2021 November | 67 | 43 | 110 |
| 2021 October | 94 | 59 | 153 |
| 2021 September | 50 | 53 | 103 |
| 2021 August | 65 | 26 | 91 |
| 2021 July | 66 | 47 | 113 |
| 2021 June | 75 | 47 | 122 |
| 2021 May | 78 | 68 | 146 |
| 2021 April | 151 | 96 | 247 |
| 2021 March | 57 | 28 | 85 |
| 2021 February | 51 | 28 | 79 |
| 2021 January | 53 | 21 | 74 |
| 2020 December | 56 | 17 | 73 |
| 2020 November | 47 | 22 | 69 |
| 2020 October | 48 | 17 | 65 |
| 2020 September | 43 | 8 | 51 |
| 2020 August | 34 | 15 | 49 |
| 2020 July | 55 | 21 | 76 |
| 2020 June | 46 | 12 | 58 |
| 2020 May | 46 | 11 | 57 |
| 2020 April | 73 | 25 | 98 |
| 2020 March | 49 | 20 | 69 |
| 2020 February | 70 | 22 | 92 |
| 2020 January | 60 | 24 | 84 |
| 2019 December | 75 | 19 | 94 |
| 2019 November | 69 | 17 | 86 |
| 2019 October | 52 | 11 | 63 |
| 2019 September | 51 | 6 | 57 |
| 2019 August | 58 | 21 | 79 |
| 2019 July | 57 | 23 | 80 |
| 2019 June | 35 | 18 | 53 |
| 2019 May | 45 | 21 | 66 |
| 2019 April | 65 | 52 | 117 |
| 2019 March | 63 | 22 | 85 |
| 2019 February | 38 | 10 | 48 |
| 2019 January | 35 | 16 | 51 |
| 2018 December | 51 | 25 | 76 |
| 2018 November | 94 | 31 | 125 |
| 2018 October | 127 | 26 | 153 |
| 2018 September | 54 | 9 | 63 |
| 2018 May | 46 | 1 | 47 |
| 2018 April | 55 | 7 | 62 |
| 2018 March | 63 | 7 | 70 |
| 2018 February | 54 | 11 | 65 |
| 2018 January | 121 | 4 | 125 |
| 2017 December | 33 | 5 | 38 |
| 2017 November | 25 | 9 | 34 |
| 2017 October | 22 | 9 | 31 |
| 2017 September | 24 | 12 | 36 |
| 2017 August | 25 | 16 | 41 |
| 2017 July | 25 | 14 | 39 |
| 2017 June | 42 | 35 | 77 |
| 2017 May | 1 | 2 | 3 |
| 2017 April | 0 | 1 | 1 |
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