Letter to the Editor
Eosinophilic granulomatosis with polyangiitis: A rare case of central airway stenosis with normal eosinophil levels
Granulomatosis eosinofílica con poliangitis: un caso de estenosis de la vía aérea central con niveles normales de eosinófilos
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(A) Bronchoscope showed obvious granulation tissue hyperplasia (red arrow) and central airway stenosis. (B) Central airway biopsy (hematoxylin–eosin staining) showed that eosinophil infiltration in blood vessels and tissues was very obvious (300×, eosinophils are shown by red arrows).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Eosinophilic granulomatosis with polyangiitis (EGPA) is a rare vasculitis, which usually manifests as eosinophilia and damage to respiratory system, urinary system and circulatory system. Because EGPA can involve many tissues in the whole body, other special manifestations may appear, which usually leads to mistakes in diagnosis and treatment. This paper reports a special case of EGPA. The patient suffered from acute asphyxia due to the stenosis of central airway area I<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">1</span></a> caused by EGPA, and the eosinophil laboratory count was normal. After emergency local interventional therapy, the patient's symptoms were relieved.</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 54-year-old male patient came to our respiratory center and complained of severe breath holding, cough and excessive phlegm. His medical history includes recurrent asthma, dyspnea and recurrent low fever within 3 years, long-term use of inhaled β-2 receptor agonist and oral prednisone acetate tablets of 10<span class="elsevierStyleHsp" style=""></span>mg. Laboratory examination 3 years ago: EOS%: 29.6%, EOS: 2.66<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleSup">9</span>/L.</p><p id="par0015" class="elsevierStylePara elsevierViewall">During the physical examination, the auscultation of both lungs showed that the breathing sounds of both lungs were low, and the vital signs showed that the body temperature was 37.5<span class="elsevierStyleHsp" style=""></span>°C. After oxygen inhalation, the oxygen saturation was only 92%, and nothing else was abnormal. Computed tomography showed airway stenosis, lung infiltration and consolidation of both lungs. The patient underwent emergency bronchoscopy, and the results showed severe glottic edema and stenosis. Granulation hyperplasia and stenosis were observed in the central airway (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A) and right main bronchus. In order to relieve the patient's asphyxia, we performed balloon dilation of airway, inflated a 10-30<span class="elsevierStyleHsp" style=""></span>mm micro-technical balloon to a pressure of 2<span class="elsevierStyleHsp" style=""></span>bar for 30<span class="elsevierStyleHsp" style=""></span>s, and performed biopsy on airway granuloma. Although this intervention relieved the asphyxia, the patient's symptoms still existed.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Further laboratory examination showed that the patient's urine routine and blood routine were normal, and the white blood cell: 9.04<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleSup">9</span>/L eosinophil: 0.04<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleSup">9</span>/L. Antibody detection showed that both C-ANCA and PR-3 were negative, and the histological evaluation of granuloma showed that submucosal granuloma was formed, and eosinophils aggregated after amplification (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>B). Combined with the past history of eosinophilia, imaging manifestations, pathological eosinophilic aggregation and objective evidence of airway granulation obstruction, we are more inclined to diagnose EGPA. The patient began to receive a high dose of glucocorticoids of 70<span class="elsevierStyleHsp" style=""></span>mg per day, and cyclophosphamide (0.6<span class="elsevierStyleHsp" style=""></span>g/m<span class="elsevierStyleSup">2</span> per pulse) was injected once. After evaluating the patients, we thought it is necessary to carry out local shovel cutting to quickly relieve suffocation, and inject triamcinolone acetonide 20<span class="elsevierStyleHsp" style=""></span>mg near two granulomas in the airway respectively. The patient's wheezing symptoms disappeared, and his cough and expectoration were significantly reduced. Two weeks later, we injected cyclophosphamide again, and gradually reduced the dosage of glucocorticoid to 20<span class="elsevierStyleHsp" style=""></span>mg per day, and then to 10<span class="elsevierStyleHsp" style=""></span>mg per day after 4 weeks. After two months of follow-up, the patient didn’t suffer from wheezing again.</p><p id="par0025" class="elsevierStylePara elsevierViewall">EGPA is a rare autoimmune disease, which usually involves the lungs. The main symptoms are asthma and cough, with pathological infiltration of lower respiratory tract, ground glass shadow and thickening of bronchial wall.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">2</span></a> However, this patient showed the central airway stenosis and normal eosinophils caused by granuloma, which is rare. Interestingly, the patient's eosinophils did not increase, and granuloma still progressed. This may be related to the long-term use of glucocorticoids. Many literatures suggest that the false normal of eosinophils will be secondary to the extensive use of glucocorticoids.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">2,3</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Regarding the treatment of EGPA, in addition to the use of glucocorticoids and cyclophosphamide recommended by the guidelines,<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">4</span></a> we have carried out local interventional therapy. This is because of the particularity of granuloma in the central airway, the patient has already appeared asphyxia symptoms. After evaluation, we think that emergency bronchial intervention is necessary. Bronchoscope shovel cutting and hormone injection have achieved good results. We followed up for 2 months and the patient did not develop wheezing symptoms.</p><p id="par0035" class="elsevierStylePara elsevierViewall">We report a case of EGPA with normal eosinophils and central airway stenosis. Its special clinical features will mislead doctors’ judgment. Past medical history and pathological results play an important role in diagnosis. Bronchoscopic interventional therapy has a good effect on acute airway stenosis, but the long-term benefit of the disease needs further study.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Authors’ contributions</span><p id="par0040" class="elsevierStylePara elsevierViewall">Li Long-zhao: Design and draft articles, and get the approval of the final version.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Sun Kai: Organize medical records, consult materials and make key revisions.</p><p id="par0050" class="elsevierStylePara elsevierViewall">Wang Hong-wu: Consent to assume responsibility for all facets of the manuscript, offer guidance on manuscript-related matters, and ultimately be responsible for the review and approval of the manuscript.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Informed consent statement</span><p id="par0055" class="elsevierStylePara elsevierViewall">Participant was fully informed of the study and gave their informed written consent before participation. This study was conducted in accordance with the Declaration of Helsinki.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Data availability statement</span><p id="par0060" class="elsevierStylePara elsevierViewall">The data that support the findings of this study are available from the corresponding author upon reasonable request.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Consent for publication</span><p id="par0065" class="elsevierStylePara elsevierViewall">All authors agree to the publication of this article.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Ethical explanation</span><p id="par0070" class="elsevierStylePara elsevierViewall">All the procedures in this study were implemented in accordance with relevant laws and institutional guidelines, and have been approved by the corresponding institutional committees and informed consent of patients. Ethical examination and approval is approved by the Ethics Committee of Dongzhimen Hospital of Beijing University of Chinese Medicine (Project No.: 2023DZMEC-484-01), and the approval time is October 12, 2023.</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Funding</span><p id="par0075" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleGrantSponsor" id="gs1">Beijing Tongzhou District Science and Technology Plan Project</span> provided financial support and article publishing expenses, and the project number was <span class="elsevierStyleGrantNumber" refid="gs1">KJ2022CX047</span>.</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Conflict of interest</span><p id="par0080" class="elsevierStylePara elsevierViewall">None.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:8 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Authors’ contributions" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Informed consent statement" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Data availability statement" ] 3 => array:2 [ "identificador" => "sec0020" "titulo" => "Consent for publication" ] 4 => array:2 [ "identificador" => "sec0025" "titulo" => "Ethical explanation" ] 5 => array:2 [ "identificador" => "sec0030" "titulo" => "Funding" ] 6 => array:2 [ "identificador" => "sec0035" "titulo" => "Conflict of interest" ] 7 => 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" => 722 "Ancho" => 1450 "Tamanyo" => 336369 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">EGPA tracheoscopy and pathological findings of a case of central airway stenosis. (A) Bronchoscope showed obvious granulation tissue hyperplasia (red arrow) and central airway stenosis. (B) Central airway biopsy (hematoxylin–eosin staining) showed that eosinophil infiltration in blood vessels and tissues was very obvious (300×, eosinophils are shown by red arrows).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:4 [ 0 => array:3 [ "identificador" => "bib0025" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "A proposed classification system of central airway stenosis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "L. Freitag" 1 => "A. Ernst" 2 => "M. Unger" 3 => "K. Kovitz" 4 => "C.H. Marquette" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1183/09031936.00132804" "Revista" => array:6 [ "tituloSerie" => "Eur Respir J" "fecha" => "2007" "volumen" => "30" "paginaInicial" => "7" "paginaFinal" => "12" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/17392320" "web" => "Medline" ] ] ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0030" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Eosinophilic granulomatosis with polyangiitis: a review" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "J. White" 1 => "S. 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Vaglio" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:6 [ "tituloSerie" => "Rheumatology (Oxford)" "fecha" => "2020" "volumen" => "59" "numero" => "Suppl. 3" "paginaInicial" => "i84" "paginaFinal" => "i94" ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0040" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Evidence-based guideline for the diagnosis and management of eosinophilic granulomatosis with polyangiitis" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "G. Emmi" 1 => "A. Bettiol" 2 => "E. Gelain" 3 => "I.M. Bajema" 4 => "A. Berti" 5 => "S. Burns" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1038/s41584-023-00958-w" "Revista" => array:6 [ "tituloSerie" => "Nat Rev Rheumatol" "fecha" => "2023" "volumen" => "19" "paginaInicial" => "378" "paginaFinal" => "393" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/37161084" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/23870206/0000016300000005/v1_202409110904/S2387020624003711/v1_202409110904/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/23870206/0000016300000005/v1_202409110904/S2387020624003711/v1_202409110904/en/main.pdf?idApp=UINPBA00004N&text.app=https://www.elsevier.es/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2387020624003711?idApp=UINPBA00004N" ]
