Case Report
Radiological Manifestations of a Rare and Unrecognized Disease: Primary Pulmonary Lymphoma
Manifestaciones radiológicas de una rara y olvidada enfermedad: el linfoma pulmonar primario
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TAC torácico con cortes coronales con presencia de opacidades bilaterales asociadas con hemorragia alveolar. Modificada. B. Biopsia nasal con afectación de la pared vascular con presencia de vasculitis. C. Tinción inmunohistoquímica específica positiva para IgG4 (25 células plasmáticas IgG4 +<span class="elsevierStyleHsp" style=""></span> por campo de gran aumento) junto al infiltrado linfoplasmocítico e histiocitos. 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Planos axiales (C y D): opacidades atelectásicas-fibróticas bilaterales de distribución peribroncovascular, con bronquiectasias y bronquielectasias de tracción. 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(C and D) (Axial planes): bilateral peribronchovascular opacities due to atelectasis and fibrosis, and bronchiectasis and traction bronchiectasis. (E) (Axial plane) and (F) (coronal plane): alveolo-interstitial opacities with bilateral and peribronchovascular distribution that coexist with other peripheral band-like ground glass opacities.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Primary pulmonary lymphoma (PPL) accounts for less than 0.5% of primary lung neoplasms.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> Its non-specific clinical and radiological manifestations make diagnosis challenging, conferring a worse prognosis. We report a small series of cases that illustrate the most common PPL.</p><p id="par0010" class="elsevierStylePara elsevierViewall">Clinical case 1: A 70-year-old woman with low-grade fever and bilateral pulmonary infiltrates on chest computed tomography (CT) (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A and B), refractory to antibiotic and corticosteroid treatment. Surgical lung biopsy (SLB) revealed primary pulmonary diffuse large B cell lymphoma (PP-DLBCL), currently in remission after R-CHOP chemotherapy (CT) (rituximab, cyclophosphamide, doxorubicin, vincristine, and prednisone).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Clinical case 2: A 63-year-old woman with bilateral alveolar infiltrates (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>C and D) and peripheral blood expression of 2.3% kappa clonal B cells. SLB showed B cell infiltration, while marginal zone B cell lymphoma of mucosa-associated lymphoid tissue (MALT lymphoma) was confirmed on bone marrow biopsy. Pulmonary resection was performed and rituximab adjuvant therapy was administered.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Clinical case 3: A 37-year-old man with cough, skin rash, and alveolar opacities on CT scan (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>E and F). SLB showed Epstein–Barr virus (EBV)-positive atypical B cell infiltrate, establishing a diagnosis of grade 1 lymphomatoid granulomatosis (LG), and a wait-and-see approach was adopted.</p><p id="par0025" class="elsevierStylePara elsevierViewall">PPLs are lymphoproliferative disorders resulting from clonal proliferation of B cells, that show parenchymal and/or bronchial involvement but no extrapulmonary involvement at diagnosis or up to 3 months later. They account for less than 1% of non-Hodgkin lymphomas,<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> of which MALT lymphoma, PP-DLBCL, and LG are the most common.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a> Their etiopathogenesis is unknown, but they are associated with autoimmune diseases,<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a> infections, and EBV in LG.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a> Generally, they predominate in men in their 50s and 60s. Half of all patients (50%) with MALT lymphoma<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> are asymptomatic at diagnosis, while PP-DLBCL and LG may have a more aggressive onset involving B symptoms.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a> LG may occur with extrapulmonary involvement, the most prevalent manifestations being cutaneous, neurological and otorhinolaryngological.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a> CT is often non-specific, and the most frequent findings include multiple bilateral ground glass and/or consolidative opacities.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">2–4</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Diagnosis requires histology – the gold standard being lung biopsy obtained by CT-guided percutaneous biopsy and transbronchial biopsy, which have a sensitivity of 80% and 88%, respectively.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a> Moreover, it is crucial not to postpone SLB if results are inconclusive, since erroneous diagnoses are made in 68% of cases, causing a mean delay of up to 2 years until final diagnosis.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">The lymphoid infiltrate in MALT lymphoma is CD20-, CD19- and CD79a-positive and CD5-negative, with a Ki67 of less than 10%, while PP-DLBCL infiltrate has a Ki-67 of 40%–60%. In LG, the infiltrate shows varying proportions of EBV-positive atypical B cells, determining a disease classification of grade I to III and prognostic repercussions.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">There is no specific treatment for PPL, so therapeutic management must be individualized. Different regimens (CHOP) or monotherapy with rituximab have been shown to be effective for MALT lymphoma, and the R-CHOP regimen has become standard in patients with PP-DLBCL, with good results.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> The treatment of LG, however, depends on tumor extension and histological grade, and remains controversial.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Generally, MALT lymphomas have the best prognosis,<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a> followed by PP-DLBCL, while LG has a 5-year mortality rate of 63%–90%.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">In conclusion, the non-specific presentation of PPL is a common source of diagnostic error, so it must be included in the differential diagnosis. Immunohistochemical study is essential for early identification and treatment.</p></span>" "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" => 1784 "Ancho" => 1300 "Tamanyo" => 257842 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) (Coronal plane) and (B) (axial plane): bilateral parenchymal consolidations with peribronchovascular, peripheral, and subpleural distribution. (C and D) (Axial planes): bilateral peribronchovascular opacities due to atelectasis and fibrosis, and bronchiectasis and traction bronchiectasis. (E) (Axial plane) and (F) (coronal plane): alveolo-interstitial opacities with bilateral and peribronchovascular distribution that coexist with other peripheral band-like ground glass opacities.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0030" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Lymphoproliferative lung disorders: clinicopathological aspects" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "V. Poletti" 1 => "C. Ravaglia" 2 => "S. Tomassetti" 3 => "C. Gurioli" 4 => "G. Casoni" 5 => "S. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 1 | 1 | 2 |
| 2024 October | 62 | 17 | 79 |
| 2024 September | 68 | 12 | 80 |
| 2024 August | 74 | 34 | 108 |
| 2024 July | 43 | 16 | 59 |
| 2024 June | 61 | 22 | 83 |
| 2024 May | 62 | 22 | 84 |
| 2024 April | 39 | 17 | 56 |
| 2024 March | 50 | 13 | 63 |
| 2024 February | 41 | 24 | 65 |
| 2024 January | 32 | 20 | 52 |
| 2023 December | 82 | 26 | 108 |
| 2023 November | 65 | 19 | 84 |
| 2023 October | 101 | 34 | 135 |
| 2023 September | 55 | 40 | 95 |
| 2023 August | 45 | 32 | 77 |
| 2023 July | 47 | 23 | 70 |
| 2023 June | 48 | 20 | 68 |
| 2023 May | 75 | 15 | 90 |
| 2023 April | 81 | 35 | 116 |
| 2023 March | 69 | 24 | 93 |
| 2023 February | 56 | 23 | 79 |
| 2023 January | 53 | 30 | 83 |
| 2022 December | 76 | 25 | 101 |
| 2022 November | 73 | 19 | 92 |
| 2022 October | 56 | 37 | 93 |
| 2022 September | 51 | 41 | 92 |
| 2022 August | 47 | 38 | 85 |
| 2022 July | 45 | 52 | 97 |
| 2022 June | 76 | 68 | 144 |
| 2022 May | 50 | 21 | 71 |
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