Hypercalcemia and bulky primary adrenal non-Hodgkin lymphoma: A case report

Díaz-Roldán, Jorge; Franch-Arcas, Guzmán; Ruiz-Luque, Virgilio

doi:10.1016/j.medcli.2021.01.023

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Se aprecia masa de partes blandas perivertebral que se extiende desde C3 a C4, con señal intermedia en T2 y STIR (flechas), con compresión medular que ocasiona mielopatía (punta de flecha verde)." ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Elena Moya Sánchez, Irene Garrido Márquez, Jade García Espinosa" "autores" => array:3 [ 0 => array:2 [ "nombre" => "Elena" "apellidos" => "Moya Sánchez" ] 1 => array:2 [ "nombre" => "Irene" "apellidos" => "Garrido Márquez" ] 2 => array:2 [ "nombre" => "Jade" "apellidos" => "García Espinosa" ] ] ] ] ] "idiomaDefecto" => "es" "Traduccion" => array:1 [ "en" => array:9 [ "pii" => "S2387020621006471" "doi" => "10.1016/j.medcle.2021.01.020" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => false "ES2" => false "LATM" => false ] "gratuito" => false "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2387020621006471?idApp=UINPBA00004N" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S002577532100186X?idApp=UINPBA00004N" "url" => "/00257753/0000015700000012/v1_202112010528/S002577532100186X/v1_202112010528/es/main.assets" ] "en" => array:15 [ "idiomaDefecto" => true "cabecera" => "Letter to the Editor" "titulo" => "Hypercalcemia and bulky primary adrenal non-Hodgkin lymphoma: A case report" "tieneTextoCompleto" => true "saludo" => "Dear Editor," "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "595" "paginaFinal" => "596" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Jorge Díaz-Roldán, Guzmán Franch-Arcas, Virgilio Ruiz-Luque" "autores" => array:3 [ 0 => array:4 [ "nombre" => "Jorge" "apellidos" => "Díaz-Roldán" "email" => array:1 [ 0 => "jorgediazcirujano@gmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "Guzmán" "apellidos" => "Franch-Arcas" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "b" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "Virgilio" "apellidos" => "Ruiz-Luque" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "c" "identificador" => "aff0015" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "Department of General and Gastrointestinal Surgery. Hospital Universitario Virgen de Valme, Sevilla, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Department of General and Gastrointestinal Surgery. Complejo Asistencial Universitario de Salamanca, Institute for Biomedical Research of Salamanca (IBSAL), Salamanca, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Department of General and Digestive Surgery. Hospital Universitario Virgen de Valme, Sevilla, Spain" "etiqueta" => "c" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Hipercalcemia y linfoma no hodgkiniano primario suprarrenal voluminoso: descripción de un caso" ] ] "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" => 1049 "Ancho" => 2175 "Tamanyo" => 223711 ] ] "descripcion" => array:1 [ "en" => "Left: Abdominal computed tomography scan showing a giant bilateral adrenal mass (white arrows) (18 × 11 × 15 and 13 cm): coronal reconstruction. Right: Evolution of the metabolic response assessed by FGD PET-CT: from September 2019 (grey arrows) to November 2020 after 2nd line chemotherapy (R-ESHAP)." ] ] ] "textoCompleto" => "Bilateral adrenal tumors are rare. Differential diagnosis must include some conditions such as pheochromocytoma, tuberculosis, primary adrenal lymphoma, metastatic disease, non-functioning adenomas, or hyperplasias among others. We present a case with aggressive features of bilateral non-Hodgkin lymphoma and review current trends.A 57-year-old man presented with a constitutional syndrome, fatigue, anorexia, and involuntary weight loss of 20 kg in two months. He experienced no other symptoms. On admission, the physical examination didn’t reveal any significant findings. Laboratory test showed very high blood calcium levels (16 mg/dL, normal range from 8.6 to 10.4 mg/dL). Parathormone was suppressed. Levels of 25-OH vitamin D, calcitonin, and serum phosphorus were, as well, low. Cortisol, aldosterone, and metanephrine levels were normal, but 1,25-OH vitamin D was high. Lactate dehydrogenase (LDH), C-reactive protein (CRP), ferritine, and β2 microglobulin were elevated.The thoracoabdominal computed tomography (CT) scan revealed giant adrenal bilateral mass (18 × 11 × 15 and 13 cm) (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>), PET-CT scan only found adrenal involvement Pathological examination, by CT-guided core biopsy, revealed large neoplastic B-cells arranging in a diffuse pattern with expression of CD20 +, BCL2 + (100%), BCL6 + (80%), MUM1 + (90%), C-MYC (>40%), CD5+, Ki67 proliferative index was 85%. Pathologic diagnosis was non-germinal center (non-GC) phenotype of diffuse large B-cell lymphoma.<elsevierMultimedia ident="fig0005"></elsevierMultimedia>Treatment was chemotherapy: R-mega-CHOP, followed by high-dose methotrexate. Adrenal bulky masses reduced volume (60%), then he presented a symptomatic mass in the thoracic wall, so he received second-line treatment with R-ESHAP. The complete response, sixteen months after diagnosis, has been then achieved and assessed by FGD PET-CT (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). Furthermore, the patient is proposed for Autologous Stem-Cell transplantation (TASP).Non-Hodgkin lymphomas (NHL) are among the top ten causes of cancer mortality, when excluding non-melanoma of skin and mixing colon and rectum cancer. Only 4% of patients with NHL showed evidence of adrenal involvement and only 1-2% at the time of the first presentation. Our patient presented some features suggesting primary adrenal lymphoma (PAL): bulky mass, constitutional syndrome and a main “very high” hypercalcemia. Typically presented an inflammatory syndrome with high levels of CRP, ferritin, LDH and β2 microglobulin as previously reported in PAL,<a class="elsevierStyleCrossRef" href="#bib0030">1</a> but didn’t show adrenal insufficiency which seems to be an usual lab test finding.<a class="elsevierStyleCrossRefs" href="#bib0030">1,2</a>A recent review only found 6 cases of hypercalcemia out of 126 patients with PAL.<a class="elsevierStyleCrossRef" href="#bib0040">3</a> In our case, according to previous report there was elevation of calcitriol (1,25-OH vitamin D).<a class="elsevierStyleCrossRef" href="#bib0040">3</a>The prognosis of PAL related to hypercalcemia is poor, that review didn’t found any survival over 12 months. In general, PAL has a poor prognosis, a systematic review of all reported cases, only found available outcomes of 149 cases, with a 12 months survival of only 20%.<a class="elsevierStyleCrossRef" href="#bib0035">2</a> Neither this review nor a newer one with 28 cases,<a class="elsevierStyleCrossRef" href="#bib0030">1</a> mentioned the relationship between hypercalcemia and PAL, adding special interest to our case who is alive with aggressive chemotherapy treatment after 16 months from diagnosis. This is the longest survival that we have found.The Ki-67 was over 80%, which has been related to a poor prognosis in NHL, with a 1- and 2-year survival rates of 60 and 0% respectively.The WHO classification, revised in 2017, placed lymphomas with concurrent MYC, BCL2, and BCL6 positive translocations, like ours (also known as triple hit lymphomas) in a new category designated as high-grade B cell lymphoma.<a class="elsevierStyleCrossRef" href="#bib0045">4</a> Furthermore, MUM1 positivity is also a predictive marker which trends to a shorter overall survival. All these features together suggest a poor prognosis in this case.The addition of Rituximab to classic CHOP and high dose (mega) was used with partial response. Some authors have proposed surgery followed by chemotherapy even there is no evidence of survival benefit. Relapse in the thoracic wall was treated with R-ESHAP therapy following current trends.<a class="elsevierStyleCrossRef" href="#bib0050">5</a>In conclusion the adrenal involvement in NHL as a first presentation is unusual and there is a lack of evidence of management. Even with a poor prognosis, every effort should be done to treat them with curative intention, including lines of chemotherapy treatment when inadequate response. Due to this low incidence, more studies should be considered in order to provide consistent evidence.Conflicts of interestThe authors have no conflicts of interest to disclose." "textoCompletoSecciones" => array:1 [ "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Conflicts of interest" ] 1 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "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" => 1049 "Ancho" => 2175 "Tamanyo" => 223711 ] ] "descripcion" => array:1 [ "en" => "Left: Abdominal computed tomography scan showing a giant bilateral adrenal mass (white arrows) (18 × 11 × 15 and 13 cm): coronal reconstruction. 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Hameed" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.21315/mjms2016.23.6.13" "Revista" => array:5 [ "tituloSerie" => "Malaysian J Med Sci" "fecha" => "2016" "volumen" => "23" "paginaInicial" => "118" "paginaFinal" => "122" ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0045" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "MYC/BCL2/BCL6 triple hit lymphoma: a study of 40 patients with a comparison to MYC/BCL2 and MYC/BCL6 double hit lymphomas" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "W. Huang" 1 => "L.J. Medeiros" 2 => "P. Lin" 3 => "W. Wang" 4 => "G. Tang" 5 => "J. Khoury" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1038/s41379-018-0067-x" "Revista" => array:7 [ "tituloSerie" => "Mod Pathol" "fecha" => "2018" "volumen" => "31" "paginaInicial" => "1470" "paginaFinal" => "1478" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/29785017" "web" => "Medline" ] ] "itemHostRev" => array:3 [ "pii" => "S0140673600042586" "estado" => "S300" "issn" => "01406736" ] ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0050" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Results of R-ESHAP as salvage therapy in refractory/relapsed follicular lymphoma: a real-world experience on behalf of GELCAB group" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "A. Muntañola" 1 => "T. Baumann" 2 => "A.C. Caballero" 3 => "B. Sánchez-González" 4 => "S. Mercadal" 5 => "L. Escoda" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1007/s00277-020-04101-7" "Revista" => array:5 [ "tituloSerie" => "Ann Hematol" "fecha" => "2020" "volumen" => "99" "paginaInicial" => "1627" "paginaFinal" => "1634" ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/00257753/0000015700000012/v1_202112010528/S0025775321001858/v1_202112010528/en/main.assets" "Apartado" => array:4 [ "identificador" => "66430" "tipo" => "SECCION" "es" => array:2 [ "titulo" => "Cartas al Editor" "idiomaDefecto" => true ] "idiomaDefecto" => "es" ] "PDF" => "https://static.elsevier.es/multimedia/00257753/0000015700000012/v1_202112010528/S0025775321001858/v1_202112010528/en/main.pdf?idApp=UINPBA00004N&text.app=https://www.elsevier.es/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0025775321001858?idApp=UINPBA00004N"]

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