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It is defined as a generalised respiratory tract neuroendocrine cell hyperplasia. Radiologically characterised by multiple lung nodules, it is more common in non-smoking women.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">We report the case of an 80-year-old woman with a several-year history of slow-growing pulmonary nodules, with a biopsy compatible with idiopathic diffuse endocrine cell hyperplasia.</p><p id="par0020" class="elsevierStylePara elsevierViewall">80-year-old woman with a personal history of essential hypertension, type 2 diabetes mellitus, hypercholesterolemia and established osteoporosis (vertebral fractures D6 and D8). Never smoker. Eight years earlier, an incidental MRI scan of the neck and back for suspected vertebral fracture revealed multiple millimetre-sized pulmonary nodules distributed over both lung fields, with a predominance of the left lower lobe. At that time, 18F-FDG PET-CT was performed, showing no radiopharmaceutical uptake, and regular radiological follow-up was indicated, but the patient was lost to follow-up. Currently, she was referred for follow-up after visualisation of the aforementioned nodules in the upper planes of a CT scan of the abdomen performed for another reason. In the history-taking by organs and systems, she only reported a chronic irritative cough with no other respiratory symptoms or associated constitutional symptoms.</p><p id="par0025" class="elsevierStylePara elsevierViewall">On physical examination, vital signs were normal, with a peripheral oxygen saturation (SpO<span class="elsevierStyleInf">2</span>) of 98% on room air. Absence of peripheral laterocervical, supraclavicular, axillary or inguinal lymphadenopathy. Auscultation and abdominal examination were normal, as well as the lower limb examination.</p><p id="par0030" class="elsevierStylePara elsevierViewall">With regard to ancillary tests, the following laboratory determinations were performed: biochemistry and hematimetry (without abnormalities), erythrocyte sedimentation rate (17 mm/h), IGRA (<span class="elsevierStyleItalic">Interferon Gamma Release Assay</span>; negative), lung tumour markers (Cyfra 21.1 and CEA in range; elevated pro-GRP 1721-normal values <50 pg/mL), antinuclear and anti-neutrophil cytoplasmic antibodies (negative). 18F-FDG PET-CT was requested and showed multiple bilateral lung nodules with slight growth compared to the test performed 8 years earlier, with a greater number and size in the left lower lobe, the largest reaching 15 mm and SUVmax 3.1. Given the slow progression of the lesions, together with the elevation of pro-GRP, the possibility of a neuroendocrine lineage was raised, so the study was completed with a PET/CT with 68Ga-DOTATOC, with the finding of multiple foci of mild/moderate uptake in lung parenchyma (SUVmax 2.4), predominantly in the left posterobasal segment, compatible with weak expression of somatostatin receptors. Both neuron-specific enolase levels in serum and 5-hydroxyindoleacetic acid levels in urine were normal.</p><p id="par0035" class="elsevierStylePara elsevierViewall">A CT-guided core needle biopsy (CNB) of the largest nodule in the left lower lobe was performed. Pathology reported foci of neuroendocrine cell proliferation with the following immunohistochemical profile: chromogranin, synaptophysin, INSM1, TTF-1, and CK AE1/AE3-positive. Absence of necrosis and/or mitosis and cell proliferation index (Ki67) <1%. All compatible with diffuse idiopathic neuroendocrine cell hyperplasia.</p><p id="par0040" class="elsevierStylePara elsevierViewall">In view of the slow progression and growth of the lesions and the very low rate of cell proliferation, clinical and radiological follow-up was decided.</p><p id="par0045" class="elsevierStylePara elsevierViewall">As mentioned above, idiopathic diffuse hyperplasia is an uncommon part of the spectrum of neuroendocrine cell proliferation, but it may predispose to synchronous carcinoid tumours and even coexist with them.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Although sometimes they are detected incidentally, about half of all cases are associated with non-specific respiratory symptoms such as dry cough, exertional dyspnoea or wheezing.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Histologically, it occurs as a generalised proliferation of neuroendocrine cells, either nodular or linear, confined to the bronchial epithelium. In most cases the course is indolent and favourable with radiological stability, although up to 30% may show progression.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Clinically, it will depend primarily on the degree of airway obstruction. However, given its status as precursor to carcinoid tumours, long-term clinical and radiological follow-up is recommended. The use of somatostatin analogues for the control of respiratory symptomatology is an option, although there are limited data.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Ethical considerations</span><p id="par0050" class="elsevierStylePara elsevierViewall">Informed consent was obtained for subsequent publication.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Funding</span><p id="par0055" class="elsevierStylePara elsevierViewall">Not funded.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflict of interest</span><p id="par0060" class="elsevierStylePara elsevierViewall">No conflict of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Ethical considerations" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Funding" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Conflict of interest" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "A multidisciplinary approach to the work up and management of pulmonary carcinoid tumors and DIPNECH: a narrative review" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "R.A. Ramirez" 1 => "A.S. Cass" 2 => "S. Das" 3 => "S.W. Low" 4 => "M. Mehrad" 5 => "O.B. Rickman" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.21037/tlcr-22-415" "Revista" => array:6 [ "tituloSerie" => "Transl Lung Cancer Res." "fecha" => "2022" "volumen" => "11" "paginaInicial" => "2567" "paginaFinal" => "2587" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/36636417" "web" => "Medline" ] ] ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0010" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Idiopathic diffuse hyperplasia of pulmonary neuroendocrine cells and airways disease" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "S.M. Aguayo" 1 => "Y.E. Miller" 2 => "J.A. Waldron Jr" 3 => "R.M. Bogin" 4 => "M.E. Sunday" 5 => "G.W. Staton Jr" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:5 [ "tituloSerie" => "N Eng J Med." 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"fecha" => "2022" "volumen" => "82" "paginaInicial" => "154" "paginaFinal" => "158" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/35037876" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/23870206/0000016300000006/v1_202409251214/S2387020624003899/v1_202409251214/en/main.assets" "Apartado" => array:4 [ "identificador" => "43309" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Letters to the Editor" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/23870206/0000016300000006/v1_202409251214/S2387020624003899/v1_202409251214/en/main.pdf?idApp=UINPBA00004N&text.app=https://www.elsevier.es/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2387020624003899?idApp=UINPBA00004N" ]
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Vol. 163. Issue 6.
Pages 310-311 (September 2024)
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Vol. 163. Issue 6.
Pages 310-311 (September 2024)
Letter to the Editor
Diffuse idiopathic pulmonary neuroendocrine cell hiperplasia: a rare cause of lung nodules
Hiperplasia difusa idiopática de células neuroendocrinas de pulmón: una causa infrecuente de nódulos pulmonares
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