Scientific Letter
Bilateral pulmonary involvement. Non-Surgical options for inflammatory pseudotumor - A case report
Afectación pulmonar bilateral. Opciones no quirúrgicas del pseudotumor inflamatorio, a propósito de un caso
José Antonio Delgado Torralbo
, María Pavón Masa, Estefanía Luque Crespo
Corresponding author
Hospital Universitario Virgen Macarena, Sevilla, Spain
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Non-Surgical options for inflammatory pseudotumor - A case report" "tieneTextoCompleto" => true "saludo" => "To the Editor:" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "49" "paginaFinal" => "50" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "José Antonio Delgado Torralbo, María Pavón Masa, Estefanía Luque Crespo" "autores" => array:3 [ 0 => array:4 [ "nombre" => "José Antonio" "apellidos" => "Delgado Torralbo" "email" => array:1 [ 0 => "joseantoniodelgadotorralbo@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "María Pavón" "apellidos" => "Masa" ] 2 => array:2 [ "nombre" => "Estefanía Luque" "apellidos" => "Crespo" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Hospital Universitario Virgen Macarena, Sevilla, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Afectación pulmonar bilateral. Opciones no quirúrgicas del pseudotumor inflamatorio, a propósito de un caso" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 2032 "Ancho" => 2500 "Tamanyo" => 321599 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Computed axial tomography image at diagnosis (A) and after treatment with corticosteroids (B), showing remission of the lesion in the right upper lobe, with some remnants of residual fibrous tracts (arrows). PET-CT image with enhanced uptake in the left pulmonary hilum (C) and in the left upper lobe (D), marked by arrows, reflecting the multilocular involvement of this case.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Inflammatory pseudotumor is a rare entity that represents less than 1 % of all tumors of the lung.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Its radiological characteristics when viewed with standard tomographic techniques overlap with those of other entities,<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> complicating differential diagnosis, especially in lung cancer.</p><p id="par0010" class="elsevierStylePara elsevierViewall">We report the case of a 49-year-old woman, former smoker with a pack-year index of 2, and a history of hypothyroidism. Her regular treatment was levothyroxine 75 mcg, 1 tablet a day. She attended the emergency department due to headache following an episode of sinusitis, with no associated respiratory symptoms. The baseline study included a standard chest X-ray, which revealed an incidental finding of a right apical lesion of irregular contours. An initial CT scan was requested that revealed a “rounded tumor mass in the right lung vertex, irregular contours, lobulated and spiculated towards the surrounding parenchyma. Lesion with heterogeneous enhancement with contrast, in extended contact with the posterior and superior apical pleura, measuring about 45<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>42<span class="elsevierStyleHsp" style=""></span>mm, with no evidence of pleural or extrapleural infiltration or extension; at the apex of the left lung, a small, nodular subpleural, lesion measuring less than 1<span class="elsevierStyleHsp" style=""></span>cm of dubious significance is observed”. In light of these findings, positron emission tomography was performed. This highlighted the presence of this lesion, which was metabolically active with a maximum standardized uptake value (standarized SUVmax) of 27.5, and the lesion in the left upper lobe, which had an SUVmax of 7.5. Bilateral hilar adenopathies were seen in the mediastinum, in particular a left hilar conglomerate measuring 3.2<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>2.1<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>4.1<span class="elsevierStyleHsp" style=""></span>cm with a SUVmax of 14.1 (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). Given the high suspicion of malignancy, we decided to conduct a core needle biopsy of the larger lesion, the result of which showed the presence of fibrosis and polymorphous lymphohistiocytic exudate suggestive of an inflammatory process or inflammatory pseudotumor. The molecular study was negative for EGFR gene mutations, but genetic studies were not performed for ALK. Tumor markers (CEA, CA 15.3, CA 19.9, CYFRA-21, squamous cell carcinoma Ag, Pro-GAP, and enolase) were negative. No anemia or thrombocytosis was detected. These results needed to be confirmed by a repeat core needle biopsy, which provided a similar diagnosis. We also performed fiberoptic bronchoscopy, which revealed mucosal thickening of inflammatory appearance in the left lower lobe. Blind bronchial brushing was performed of this region, yielding no neoplastic cells. In the absence of any suggestion of malignancy, and the existence of several lesions that prevented a surgical approach, we decided to start treatment with deflazacor at doses of 1<span class="elsevierStyleHsp" style=""></span>mg/kg body weight per day for 2 months, tapering to half in the following 2 months, and discontinuing the following month. Three computed axial tomography scans were performed during treatment, showing a progressive decrease in tumor size, as well as the presence of linear tracts of fibrotic scar tissue and traction bronchiectasis in the area, with disappearance of the contralateral nodule.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Inflammatory pseudotumor can occur in any organ, but it mainly affects the lung<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> and occurs more frequently in the first 2 decades of life.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> It usually presents as a single, circumscribed mass measuring more than 3<span class="elsevierStyleHsp" style=""></span>cm, and is more common in the lower lobes.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> It has been described in the literature under different names (plasma cell granuloma, inflammatory myofibroblastic tumor or proliferation, xanthoma, fibroxanthoma, xanthogranuloma, fibrous xanthoma, xanthomatous pseudotumor, plasmocytoma, solitary mast cell granuloma, inflammatory fibrosarcoma),<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> bearing witness to the complexity, histological heterogeneity, and variable behavior of this entity that, at times, can even be classified as a true neoplasm.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Its etiology is unknown, although there are believed to be several predisposing factors: surgeries, trauma, immune reactions, steroids, radiation therapy, and infections.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> The role of an infectious agent seems to be restricted to the early stages, when it triggers a cascade of reactions, through which the tumor develops autonomy.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> The microorganisms most frequently associated with inflammatory pseudotumor include mycobacteria, Epstein-Barr virus, <span class="elsevierStyleItalic">Actinomycetos</span> and <span class="elsevierStyleItalic">Mycoplasma</span>, and some cases have been reported in association with <span class="elsevierStyleItalic">Corynebacterium equi, Escherichia coli, Klebsiella, Bacillus sphaericus, Pseudomonas, Helicobacter pylori, Coxiella burnetii</span>,<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,8,9</span></a> herpes simplex virus, and even with HIV infection.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> Pathological study shows fibrous bands interspersed by irregular layers of lymphocytes, histiocytes, and polyclonal plasma cells.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">11</span></a> Although surgical resection is the treatment of choice,<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">12</span></a> several recent studies have reported rearrangements of the ALK gene on chromosome 2p23 causing aberrant expression in about 50 % of cases.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">13</span></a> This suggests that this subgroup could show sensitivity to tyrosine kinase inhibitors such as crizotinib.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">14</span></a> The only publications supporting their effectiveness are case series,<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6,13</span></a> and no trials have been conducted to compare the administration of crizotinib compared to corticosteroids in this subgroup.</p><p id="par0020" class="elsevierStylePara elsevierViewall">The efficacy of corticosteroid therapy varies, and the results reported in the literature<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,12,14,15</span></a> from first-line use in patients who are not candidates for surgery<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,14,15</span></a> (e.g., bilateral presentation) are limited and inconclusive. Lee et al. reported a similar case in which the response to corticosteroids was excellent, showing no evidence of recurrence after 20 months of follow-up after disappearance of the lesions.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">14</span></a> Díez et al. obtained similar results after a 30-month course of low-dose corticosteroid therapy.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">15</span></a> Few cases of bilateral lesions, such as those presented in our patient, have been reported, and the effectiveness and recurrence rate after steroid treatment are unknown, due to the absence of studies in this area.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2019-04-27" "fechaAceptado" => "2019-06-06" "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Delgado Torralbo JA, Masa MP, Crespo EL. Afectación pulmonar bilateral. Opciones no quirúrgicas del pseudotumor inflamatorio, a propósito de un caso. Arch Bronconeumol. 2020;56:49–51.</p>" ] ] "multimedia" => array:1 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 2032 "Ancho" => 2500 "Tamanyo" => 321599 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Computed axial tomography image at diagnosis (A) and after treatment with corticosteroids (B), showing remission of the lesion in the right upper lobe, with some remnants of residual fibrous tracts (arrows). PET-CT image with enhanced uptake in the left pulmonary hilum (C) and in the left upper lobe (D), marked by arrows, reflecting the multilocular involvement of this case.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:15 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "[1]" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Pulmonary inflammatory pseudotumor causing collapse responding to corticosteroid therapy" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "R.Z. Reddy" 1 => "Y.M. Carter" 2 => "D.W. Hsia" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:7 [ "tituloSerie" => "Resp Med Case Rep." 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| Year/Month | Html | Total | |
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| 2024 November | 2 | 1 | 3 |
| 2024 October | 55 | 31 | 86 |
| 2024 September | 55 | 18 | 73 |
| 2024 August | 62 | 37 | 99 |
| 2024 July | 40 | 21 | 61 |
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| 2024 May | 55 | 46 | 101 |
| 2024 April | 33 | 27 | 60 |
| 2024 March | 41 | 15 | 56 |
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| 2023 March | 8 | 3 | 11 |
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| 2022 December | 56 | 33 | 89 |
| 2022 November | 66 | 23 | 89 |
| 2022 October | 50 | 36 | 86 |
| 2022 September | 35 | 12 | 47 |
| 2022 August | 34 | 46 | 80 |
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| 2022 June | 46 | 40 | 86 |
| 2022 May | 40 | 36 | 76 |
| 2022 April | 31 | 35 | 66 |
| 2022 March | 37 | 37 | 74 |
| 2022 February | 33 | 35 | 68 |
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| 2020 May | 1 | 0 | 1 |
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