Pediatric Pulmonology 47:447–452 (2012) Bronchiectasis in Chronic Pulmonary Aspiration: Risk Factors and Clinical Implications 1 1 2 Joseph C. Piccione, DO, MS, Gary L. McPhail, MD, Matthew C. Fenchel, MS, 3 4 Alan S. Brody, MD, and Richard P. Boesch, DO, MS * Summary. Introduction: Bronchiectasis is a well-known sequela of chronic pulmonary aspira- tion (CPA) that can result in significant respiratory morbidity and death. However, its true preva- lence is unknown because diagnosis requires high resolution computed tomography which is not routinely utilized in this population. This study describes the prevalence, time course for development, and risk factors for bronchiectasis in children with CPA. Materials and Methods: Using a cross-sectional design, medical records were reviewed for all patients with swallow study or airway endoscopy-confirmed aspiration in our airway center over a 21 month period. All patients underwent rigid and flexible bronchoscopy, and high resolution chest computed tomography. Prevalence, distribution, and risk factors for bronchiectasis were identified. Results: One hundred subjects age 6 months to 19 years were identified. Overall, 66% had bronchiectasis, including 51% of those less than 2 years old. The youngest was 8 months old. Severe neurological impairment (OR 9.45, P < 0.004) and history of gastroesophageal reflux (OR 3.36, P ¼ 0.036) were identified as risk factors. Clinical history, exam, and other co-mor- bidities did not predict bronchiectasis. Sixteen subjects with bronchiectasis had repeat chest computed tomography with 44% demonstrating improvement or resolution. Discussion: Bron- chiectasis is highly prevalent in children with CPA and its presence in young children demon- strates that it can develop rapidly. Early identification of bronchiectasis, along with interventions aimed at preventing further airway damage, may minimize morbidity and mortality in patients with CPA. Pediatr Pulmonol. 2012; 47:447–452. ß 2011 Wiley Periodicals, Inc. Key words: aspiration pneumonia; deglutition disorders; children. Funding source: none reported. INTRODUCTION 1Division of Pulmonary Medicine, Cincinnati Children’s Hospital Medi- cal Center, Cincinnati, Ohio. Bronchiectasis is the abnormal dilatation of bronchi and bronchioles that results from damage to the airways 2Division of Biostatistics and Epidemiology, Cincinnati Children’s Hospi- caused by infection and inflammation.1,2 It can lead to tal Medical Center, Cincinnati, Ohio. considerable morbidity including chronic cough, recur- 3 rent infections, and impaired lung function.3 When Department of Radiology, Cincinnati Children’s Hospital Medical Center, Cincinnati, Ohio. bronchiectasis develops during childhood it may be- come irreversible and lead to chronic disease into adult- 4Division of Pulmonary Medicine and Aerodigestive & Sleep Center, hood with increased risk for mortality due to Cincinnati Children’s Hospital Medical Center, Cincinnati, Ohio. respiratory failure.2,4 The current gold standard for diagnosis of bronchiec- Conflicts of interest: None. tasis is high resolution chest CT (HRCT). HRCT is a An earlier version of this manuscript was published in abstract form and radiographic technique ideally suited to evaluate diffuse presented in slide format at the American College of Chest Physicians lung diseases and the images obtained are representa- Meeting in November, 2009. tive of those found on histopathology.5 HRCT uses lim- ited sampling which allows radiation doses to be *Correspondence to: Richard P. Boesch, DO, MS, Division of Pulmonary Medicine, Cincinnati Children’s Hospital Medical Center, MLC 2021, minimized and current protocols deliver doses equiva- 3333 Burnet Avenue, Cincinnati, 45229 Ohio. 6 lent to 6 weeks of ambient radiation exposure. E-mail: [email protected] Chronic pulmonary aspiration (CPA) is a well-known cause of bronchiectasis in children, but several recent Received 14 June 2011; Accepted 23 September 2011. reviews of non-cystic fibrosis bronchiectasis implicate 7–11 DOI 10.1002/ppul.21587 CPA as the cause in only 2–18% of cases. Because Published online 25 October 2011 in Wiley Online Library HRCT is not routinely utilized in the evaluation and (wileyonlinelibrary.com). ß 2011 Wiley Periodicals, Inc. 448 Piccione et al. management of children with CPA, this cause of bron- patients diagnosed with CPA after evaluation in this chiectasis may be underrepresented in these studies. center between July 2006 and March 2009. Chronic pulmonary aspiration is characterized by the recurrent spillage of food, regurgitated material due to Diagnostic Evaluation gastro-esophageal reflux, and/or oral secretions into All patients underwent rigid and flexible bronchosco- the subglottic airways.12 Causes include dysfunctional py, HRCT of the chest, and at least one swallowing swallowing, impaired airway protective responses, study at the time of their initial evaluation. The diagno- and abnormal anatomical connections between the air- sis of CPA was made if the swallowing study (VSS or way and gastrointestinal tract. CPA is the leading FEES) demonstrated aspiration, or if either a laryngeal cause of death in children with severe neurological cleft (excluding type I cleft) or TEF was identified on disorders.13–15 Clinical symptoms are non-specific, in- endoscopic examination. Chest HRCT was performed cluding chronic cough, wheezing, and recurrent pneu- using controlled ventilation techniques with thin monia.12,16 When these symptoms are caused by other (1 mm) images obtained at intervals of 10 mm during a conditions, however, bronchiectasis is not an expected controlled inspiratory breath hold and then again at finding.17 20 mm intervals in exhalation. Images were reviewed Video fluoroscopic swallowing studies (VSS) and for the presence and location of bronchiectasis as speci- fiberoptic-endoscopic evaluation of swallowing (FEES) fied by the radiographic definition described in the are part of the diagnostic evaluation for CPA.18,19 In Fleischner Society lexicon.21 Patient characteristics in- addition, flexible bronchoscopy with bronchoalveolar cluding co-morbid conditions are described in Table 1. lavage (BAL) can be performed to obtain lower respira- Their presenting symptoms and physical exam findings tory secretions for culture and cytological analysis, and were recorded. BAL fluid analysis was reviewed to aid in the identification of anatomical abnormalities. identify markers of airway inflammation and infection Rigid bronchoscopy is the preferred method for evalua- (defined as >106 colonies/ml of a single organism on tion of suspected laryngeal cleft or tracheoesophageal bacterial culture). When available, results of esophageal fistula (TEF).20 impedance studies and any follow-up HRCT scans were The prevalence of bronchiectasis in children with also reviewed. CPA has not been well described nor is it clear if the presence of bronchiectasis is related to the mechanism Statistical Analysis of aspiration, the material aspirated, or other clinical correlates (such as: gender, prematurity, tracheostomy Summary statistics were used to describe the age at status, ventilator dependence, or severe neurological identification of bronchiectasis and the extent and ana- disease). The aim of this study is to describe the preva- tomical distribution of bronchiectasis when present. lence, distribution, and time course for the development The frequency of respiratory symptoms, physical of bronchiectasis in children with CPA. We compare exam findings, and co-morbid conditions were com- the symptoms, physical exam findings, and lower air- pared between those with and without bronchiectasis way inflammatory markers of those with and without using Chi-square or Fisher’s exact tests (depending on bronchiectasis and attempt to identify predictors of its sample size). Age and mean percent neutrophils presence. We also investigate the role of CPA etiology and lipid laden macrophages were compared using and co-morbidities as possible risk factors for the devel- the Kruskal–Wallis non-parametric test. The proportions opment of bronchiectasis. of subjects with airway infection were compared using a Chi-Square test. Potential risk factors for the MATERIALS AND METHODS Study Population TABLE 1— Patient Characteristics The study was approved by the Institutional Review Age (mean in months) 6 month–19 year (49) Board and a waiver of informed consent was granted. The study population was selected from our Aerodiges- Gender (M, F) 56, 44 tive and Sleep Center. This program is a tertiary referral VSS positive for aspiration (%) 62 FEES positive for aspiration (%) 51 program in which children with multi-system disease Laryngotracheoesophageal cleft or TEF (%) 12 receive coordinated multi-disciplinary evaluation and Premature birth (%) 53 treatment for airway reconstruction, chronic aspiration, Severe neurological impairment (%) 30 and feeding disorders. Approximately 100 new pediatric Tracheostomy status (%) 50 patients are evaluated annually, many of whom are sus- Ventilator dependence (%) 9 Reported history of GER (%) 80 pected of having CPA as a part of their clinical spec- History of prior fundoplication (%) 56 trum. Medical records were reviewed for all new Pediatric Pulmonology Bronchiectasis in Chronic Pulmonary Aspiration 449 development of bronchiectasis within this population of of BAL fluid analysis, and co-morbid conditions be- children with CPA were evaluated by logistic regres- tween subjects with and without bronchiectasis. There sion. The initial model included: