Letter to the Editor
Hepatosplenic gammadelta T-cell lymphoma and Sjögren's syndrome
Linfoma T hepatoesplénico gamma-delta y síndrome de Sjögren
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Staub" "autores" => array:4 [ 0 => array:3 [ "nombre" => "Ronaldo" "apellidos" => "Godinho" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 1 => array:3 [ "nombre" => "Paula Vanessa" "apellidos" => "de Oliveira" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "Deonilson Ghizoni" "apellidos" => "Schmoeller" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 3 => array:4 [ "nombre" => "Henrique L." "apellidos" => "Staub" "email" => array:1 [ 0 => "reumatoacademico@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" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Rheumatology Department, Faculty of Medicine, Saint Lucas Hospital, Pontifical Catholic University of Rio Grande do Sul, Porto Alegre, Brazil" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Hematology Department, Faculty of Medicine, Saint Lucas Hospital, Pontifical Catholic University of Rio Grande do Sul, Porto Alegre, Brazil" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Linfoma T hepatoesplénico gamma-delta y síndrome de Sjögren" ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Individuals with primary Sjögren's syndrome (pSS) have over 40-fold increased risk of the development B-cell lymphoma.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> The relationship of SS with T-cell lymphoma is, nevertheless, enigmatic. We herein describe a case of a patient with features compatible with SS who evolved to a hepatosplenic gammadelta T-cell lymphoma (GDTL).</p><p id="par0010" class="elsevierStylePara elsevierViewall">The patient, a 25-year-old white female, had complained of fatigue, “dry eyes” (confirmed by an Ophthalmologist), parotid enlargement and xerostomia for the last four years. Physical examination in December 2008 revealed increased parotid glands and hepatosplenomegaly, but no peripheral lymphadenopathy. Pancytopenia was present (hemoglobin 7.9<span class="elsevierStyleHsp" style=""></span>g/dL, white blood cell 1000<span class="elsevierStyleHsp" style=""></span>cells/mm<span class="elsevierStyleSup">3</span>, platelet count 107,000<span class="elsevierStyleHsp" style=""></span>cells/mm<span class="elsevierStyleSup">3</span>). The erythrocyte sedimentation rate was of 37<span class="elsevierStyleHsp" style=""></span>mm in the first hour. Polyclonal hypergammaglobulinemia was present. The antinuclear antibody test was strongly positive (1/5120, speckled pattern), and anti-SSA antibodies were detected in high levels (124 units in an ELISA). The rheumatoid factor test was weakly positive (45 units). Abdominal ecography confirmed hepatosplenomegaly. A bone marrow biopsy (BMB) showed hypercellularity, without evidence of malignancy. Considering the clinical and laboratory findings suggestive of pSS, the patient was treated with prednisone and azathioprine.</p><p id="par0015" class="elsevierStylePara elsevierViewall">After eight months, a notable improvement of clinical and hematological features was seen. Hepatosplenomegaly remained, however, and a new BMB plus splenectomy were carried out. At that time, hemoglobin was 10<span class="elsevierStyleHsp" style=""></span>g/dL, the leukocyte count was 21,900<span class="elsevierStyleHsp" style=""></span>cells/cm<span class="elsevierStyleSup">3</span> (with 65 erythroblasts per 100 leukocytes), and the platelet count was 106,000/cm<span class="elsevierStyleSup">3</span>. The spleen histology was inconclusive, and the BMB showed interstitial infiltration by atypical lymphoid cells. Immunohistochemistry of spleen and bone marrow revealed the following lymphocyte profile: CD3+, CD4−, CD5−, CD8−, Ki 67+ with a rate of 50% CD56+ focal. The karyotype showed, of importance, eight trisomy and absence of chromosome seven. Altogether, morphologic, phenotypic and genetic findings were compatible with a hepatosplenic GDTL. After eighteen months of standard chemotherapy, the patient died in September 2011.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Hepatosplenic GDTL is an aggressive and uncommon malignancy (<1% of lymphoid neoplasms). Intense gammadelta T-cell proliferation is characteristically seen in liver, spleen and bone marrow sinusoids.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> The disorder usually affects young adults, and the outcome is poor.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Hepatosplenomegaly and severe cytopenias, both seen in our patient, are usual aspects; lymphadenopathy is rare.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Of interest, hepatosplenic GDTL can mimic the hemophagocytic syndrome.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> In 2009, a cutaneous GDTL was diagnosed in a patient with rheumatoid arthritis using etanercept.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">Current ACR classification criteria for SS include autoantibodies, ocular staining and salivary gland histology, suggesting that case definition requires two of the three.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> pSS was initially a suitable diagnosis for our patient due to the presence of ophthalmic sicca, parotiditis, typical autoantibodies, polyclonal hypergammaglobulinemia, and peripheral pancytopenia. The first BMB showed no malignancy, and clinical features responded well to traditional immunosuppression.</p><p id="par0030" class="elsevierStylePara elsevierViewall">The unexpected persistence of hepatosplenomegaly led to an immunohistochemistry diagnosis of hepatosplenic GDTL eight months ahead. Thus, it is conceivable that the patient firstly had pSS and later developed a GDTL. Although one cannot rule out the possibility that she presented GDTL with SS features since the beginning, the reported median survival time of six months for GDTL<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> turns it less plausible. Also, an atypical form of SS “secondary” to GDTL could be brought about. Necessary to say, the occurrence of SS and GDTL could have been only coincidental in our patient. For any of these circumstances, no similar clinical scenario combining SS features and GDTL has been described so far.</p><p id="par0035" class="elsevierStylePara elsevierViewall">In summary, we describe a case of a young patient with features of SS who evolved, unusually, to a hepatosplenic GDTL. The interplay of SS with non-B lymphomas has yet to be clarified.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflict of interest</span><p id="par0040" class="elsevierStylePara elsevierViewall">The authors have no conflict of interest to declare.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Conflict of interest" ] 1 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:8 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Lymphoproliferative disorders in Sjögren's syndrome" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "M. 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Bi" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.humpath.2011.01.034" "Revista" => array:6 [ "tituloSerie" => "Hum Pathol" "fecha" => "2011" "volumen" => "42" "paginaInicial" => "1965" "paginaFinal" => "1978" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/21683978" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/21735743/0000001000000004/v1_201406280041/S2173574314001063/v1_201406280041/en/main.assets" "Apartado" => array:4 [ "identificador" => "8400" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Letters to the Editor" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/21735743/0000001000000004/v1_201406280041/S2173574314001063/v1_201406280041/en/main.pdf?idApp=UINPBA00004M&text.app=https://reumatologiaclinica.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2173574314001063?idApp=UINPBA00004M" ]
| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 1 | 4 | 5 |
| 2024 October | 49 | 34 | 83 |
| 2024 September | 53 | 17 | 70 |
| 2024 August | 56 | 26 | 82 |
| 2024 July | 38 | 28 | 66 |
| 2024 June | 34 | 30 | 64 |
| 2024 May | 38 | 26 | 64 |
| 2024 April | 30 | 21 | 51 |
| 2024 March | 40 | 32 | 72 |
| 2024 February | 40 | 30 | 70 |
| 2024 January | 24 | 18 | 42 |
| 2023 December | 24 | 23 | 47 |
| 2023 November | 38 | 25 | 63 |
| 2023 October | 38 | 22 | 60 |
| 2023 September | 55 | 32 | 87 |
| 2023 August | 29 | 12 | 41 |
| 2023 July | 38 | 32 | 70 |
| 2023 June | 30 | 21 | 51 |
| 2023 May | 39 | 21 | 60 |
| 2023 April | 22 | 4 | 26 |
| 2023 March | 28 | 28 | 56 |
| 2023 February | 39 | 23 | 62 |
| 2023 January | 24 | 15 | 39 |
| 2022 December | 36 | 42 | 78 |
| 2022 November | 53 | 27 | 80 |
| 2022 October | 46 | 24 | 70 |
| 2022 September | 37 | 33 | 70 |
| 2022 August | 30 | 27 | 57 |
| 2022 July | 28 | 43 | 71 |
| 2022 June | 38 | 37 | 75 |
| 2022 May | 46 | 44 | 90 |
| 2022 April | 42 | 40 | 82 |
| 2022 March | 43 | 39 | 82 |
| 2022 February | 35 | 33 | 68 |
| 2022 January | 29 | 30 | 59 |
| 2021 December | 27 | 39 | 66 |
| 2021 November | 33 | 43 | 76 |
| 2021 October | 38 | 42 | 80 |
| 2021 September | 33 | 45 | 78 |
| 2021 August | 36 | 27 | 63 |
| 2021 July | 43 | 29 | 72 |
| 2021 June | 41 | 31 | 72 |
| 2021 May | 47 | 28 | 75 |
| 2021 April | 114 | 82 | 196 |
| 2021 March | 44 | 21 | 65 |
| 2021 February | 43 | 17 | 60 |
| 2021 January | 43 | 12 | 55 |
| 2020 December | 51 | 21 | 72 |
| 2020 November | 40 | 17 | 57 |
| 2020 October | 25 | 10 | 35 |
| 2020 September | 30 | 23 | 53 |
| 2020 August | 36 | 20 | 56 |
| 2020 July | 26 | 9 | 35 |
| 2020 June | 29 | 17 | 46 |
| 2020 May | 22 | 12 | 34 |
| 2020 April | 24 | 13 | 37 |
| 2020 March | 13 | 8 | 21 |
| 2020 February | 1 | 0 | 1 |
| 2018 December | 2 | 0 | 2 |
| 2018 May | 5 | 1 | 6 |
| 2018 April | 25 | 7 | 32 |
| 2018 March | 32 | 6 | 38 |
| 2018 February | 21 | 2 | 23 |
| 2018 January | 13 | 3 | 16 |
| 2017 December | 26 | 3 | 29 |
| 2017 November | 16 | 8 | 24 |
| 2017 October | 21 | 8 | 29 |
| 2017 September | 24 | 7 | 31 |
| 2017 August | 21 | 6 | 27 |
| 2017 July | 25 | 8 | 33 |
| 2017 June | 37 | 10 | 47 |
| 2017 May | 40 | 8 | 48 |
| 2017 April | 24 | 4 | 28 |
| 2017 March | 19 | 9 | 28 |
| 2017 February | 22 | 4 | 26 |
| 2017 January | 23 | 5 | 28 |
| 2016 December | 39 | 19 | 58 |
| 2016 November | 33 | 4 | 37 |
| 2016 October | 42 | 5 | 47 |
| 2016 September | 39 | 8 | 47 |
| 2016 August | 29 | 1 | 30 |
| 2016 July | 26 | 4 | 30 |
| 2016 May | 1 | 0 | 1 |
| 2016 April | 4 | 0 | 4 |
| 2016 March | 2 | 0 | 2 |
| 2016 January | 2 | 0 | 2 |
| 2015 December | 1 | 0 | 1 |
| 2015 November | 2 | 0 | 2 |
| 2015 October | 1 | 0 | 1 |
| 2015 August | 2 | 0 | 2 |
| 2015 July | 10 | 10 | 20 |
| 2015 June | 19 | 7 | 26 |
| 2015 May | 32 | 12 | 44 |
| 2015 April | 16 | 7 | 23 |
| 2015 March | 21 | 5 | 26 |
| 2015 February | 10 | 5 | 15 |
| 2015 January | 20 | 10 | 30 |
| 2014 December | 17 | 11 | 28 |
| 2014 November | 20 | 11 | 31 |
| 2014 October | 17 | 6 | 23 |
| 2014 September | 22 | 12 | 34 |
| 2014 August | 20 | 20 | 40 |
| 2014 July | 36 | 22 | 58 |
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