Scientific Letter
Bronchiolitis Obliterans in a Transplant-Waitlisted Patient With Hepatorenal Polycystic Disease
Bronquiolitis obliterante en paciente con poliquistosis hepatorrenal en lista para trasplante
Laura Ibáñez Beltrána,
, Jose María García Sánchezb, Isidoro Cortell Aznara, Santiago Mendizábal Oteizac
Corresponding author
a Servicio de Neumología Infantil, Hospital Universitario y Politécnico La Fe, Valencia, Spain
b Servicio de Cirugía Plástica, Estética y Reparadora, Hospital Universitario y Politécnico La Fe, Valencia, Spain
c Servicio de Nefrología Infantil, Hospital Universitario y Politécnico La Fe, Valencia, Spain
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History significant for: chronic renal impairment, left pyelostomy, arterial hypertension, hemophagocytic syndrome at the age of 3 years due to a viral infection, hepatic fibrosis, and severe hepatosplenomegaly with chronic secondary pancytopenia. He continues to present exacerbations of pancytopenia coinciding with infectious episodes, with no medullary involvement. No respiratory history of interest. At the time of admission, he had been on the waiting list for liver-kidney transplantation for 1 year.</p><p id="par0015" class="elsevierStylePara elsevierViewall">He consulted due to a febrile episode lasting 48<span class="elsevierStyleHsp" style=""></span>h, with sparsely productive cough as the only symptom. On examination, he showed good general status, asthenia, mild tachypnea, and fine crackles in the right lung base. Clinical laboratory tests showed C-reactive protein 220<span class="elsevierStyleHsp" style=""></span>mg/l and procalcitonin 7.4<span class="elsevierStyleHsp" style=""></span>ng/ml, deteriorating creatinine clearance and worsening of baseline pancytopenia. Chest radiograph was normal. With sepsis of gastrointestinal origin as an initial diagnosis, treatment began with meropenem; the patient became afebrile 24<span class="elsevierStyleHsp" style=""></span>h later and his infection parameters improved. Urinary, gastrointestinal, and ENT infectious foci and pneumonia were ruled out. Despite clinical improvement, bilateral pulmonary and migratory crackles persisted, with normal oxygen saturation at all times, so during the week that he was hospitalized, the following tests were conducted: pulmonary ventilation–perfusion scintigraphy, normal; spirometry, mixed pattern; diffusing capacity of the lung for carbon monoxide, lower limit of normal; and finally high-resolution CT (HRCT) that confirmed the suspected diagnosis of BO (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>, mosaic attenuation pattern).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">The treatment of BO is complex, complicating the management of our patient and conferring an unfavorable prognosis. On the one hand, BO contraindicated kidney-liver transplantation and, in turn, lung transplantation would probably be required to cure possibly advanced BO in the future, yet that would be ruled out by the patient's renal impairment.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> The patient was taken off the kidney-liver transplantation waitlist. Pulmonary treatment began with inhaled budesonide (800<span class="elsevierStyleHsp" style=""></span>μg/day),<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> salbutamol on demand, and azithromycin (250<span class="elsevierStyleHsp" style=""></span>mg/3 days per week). Post-infective BO was considered the most likely etiology, but this diagnosis was ruled out after polymerase chain reaction (PCR) was negative for respiratory viruses in 2 nasopharyngeal samples.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a> Chronic immunosuppression due to pancytopenia secondary to continued hypersplenism was then thought to be the likely etiology of the BO. Splenectomy was performed, and an immediate improvement in neutropenia and thrombocytopenia was observed after surgery. Anemia persisted due to the patient's chronic kidney failure. After 3 months of respiratory treatment, he was symptom-free, spirometry was normal, and HRCT showed no lesions. Given the resolution of the BO and pancytopenia, respiratory therapy was discontinued, and the patient was included in the transplantation list again. The procedure was performed 6 months later with satisfactory progress to date.</p><p id="par0025" class="elsevierStylePara elsevierViewall">BO is a clinical syndrome characterized by chronic and irreversible airflow obstruction caused by inflammatory changes associated with fibrosis in the small airway.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">2,5</span></a> The most common etiologies in children are immunosuppression and postinfective syndromes.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">1,4</span></a> The clinical picture in most patients is very nonspecific, so the initial diagnostic approach is difficult.<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">4,5</span></a> Before BO is suspected, spirometry must show a fixed obstructive pattern and the characteristic mosaic attenuation pattern (areas of air trapping and atelectasis, and bronchial dilation) must be observed on HRCT (inspiration and expiration). Biopsy is usually not required if HRCT is available.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a> The initial treatment consists of combined therapy with inhaled glucocorticoids, macrolides, and beta-2 adrenergic bronchodilators.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> When the disease is established, this treatment is supportive and must include immunosuppressive drugs. Lung transplantation is indicated in patients who have progressed.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">In our patient, respiratory therapy and splenectomy led to clinical improvement, so in the absence of other data, the most likely etiological hypothesis was chronic immunodeficiency secondary to pancytopenia that generated hypersplenism due to liver fibrosis.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Ibáñez Beltrán L, García Sánchez JM, Cortell Aznar I, Mendizábal Oteiza S. Bronquiolitis obliterante en paciente con poliquistosis hepatorrenal en lista para trasplante. Arch Bronconeumol. 2019;55:112.</p>" ] ] "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" => 442 "Ancho" => 755 "Tamanyo" => 22813 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">HRCT image in expiration showing areas of severe air trapping and multifocal, mosaic attenuation pattern.</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" => "Bronquiolitis obliterante. Evolución a medio plazo" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "O. 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M Filho" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:6 [ "tituloSerie" => "Rev Paul Pediatr" "fecha" => "2013" "volumen" => "31" "paginaInicial" => "10" "paginaFinal" => "16" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/23703038" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/15792129/0000005500000002/v1_201902020656/S1579212918304269/v1_201902020656/en/main.assets" "Apartado" => array:4 [ "identificador" => "49861" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Scientific letters" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/15792129/0000005500000002/v1_201902020656/S1579212918304269/v1_201902020656/en/main.pdf?idApp=UINPBA00003Z&text.app=https://archbronconeumol.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1579212918304269?idApp=UINPBA00003Z" ]
| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 3 | 2 | 5 |
| 2024 October | 39 | 18 | 57 |
| 2024 September | 56 | 13 | 69 |
| 2024 August | 77 | 47 | 124 |
| 2024 July | 60 | 20 | 80 |
| 2024 June | 46 | 24 | 70 |
| 2024 May | 50 | 33 | 83 |
| 2024 April | 39 | 28 | 67 |
| 2024 March | 31 | 15 | 46 |
| 2024 February | 30 | 18 | 48 |
| 2023 March | 4 | 5 | 9 |
| 2023 February | 27 | 14 | 41 |
| 2023 January | 27 | 29 | 56 |
| 2022 December | 46 | 25 | 71 |
| 2022 November | 48 | 20 | 68 |
| 2022 October | 65 | 38 | 103 |
| 2022 September | 40 | 23 | 63 |
| 2022 August | 39 | 44 | 83 |
| 2022 July | 36 | 37 | 73 |
| 2022 June | 25 | 29 | 54 |
| 2022 May | 34 | 29 | 63 |
| 2022 April | 39 | 32 | 71 |
| 2022 March | 50 | 47 | 97 |
| 2022 February | 57 | 38 | 95 |
| 2022 January | 46 | 37 | 83 |
| 2021 December | 30 | 39 | 69 |
| 2021 November | 27 | 34 | 61 |
| 2021 October | 37 | 57 | 94 |
| 2021 September | 31 | 46 | 77 |
| 2021 August | 31 | 38 | 69 |
| 2021 July | 13 | 18 | 31 |
| 2021 June | 33 | 59 | 92 |
| 2021 May | 50 | 37 | 87 |
| 2021 April | 50 | 71 | 121 |
| 2021 March | 34 | 21 | 55 |
| 2021 February | 26 | 16 | 42 |
| 2020 March | 14 | 6 | 20 |
| 2020 February | 19 | 11 | 30 |
| 2020 January | 25 | 13 | 38 |
| 2019 December | 23 | 18 | 41 |
| 2019 November | 18 | 17 | 35 |
| 2019 October | 30 | 9 | 39 |
| 2019 September | 22 | 7 | 29 |
| 2019 August | 21 | 9 | 30 |
Show all
