Unilateral glossodynia as a harbinger of an occult cerebellopontine angle tumour.

A woman in her late 80s with severe bronchomalacia was referred to a tertiary orofacial pain clinic for unexplained right unilateral glossodynia of progressive and continuous evolution for the past 8 months, spreading to the ipsilateral labiomental region, associated with ipsilateral hypoacusia. Local and general clinical examinations were unremarkable and routine blood work could not reveal any underlying systemic disease explaining the glossodynia and burning/pricking labiomental pain. Suspecting a painful trigeminal neuropathy secondary to a space-occupying lesion, a cerebral MRI was prescribed, revealing an ipsilateral cerebellopontine angle lesion, compatible with either a schwannoma or meningioma. This lesion invaded the root entry zones of cranial nerves V and VIII explaining the patient's oral pain and hypoacusia. Following a neurosurgical consultation where surgical treatment was rejected, her pain was successfully managed by topical pregabalin mouthwashes, to prevent any risk of respiratory depression related to her underlying severe bronchomalacia.

Successful repair of critical bronchial stenosis with coexisting bronchomalacia and congenital heart defects in infants by slide bronchoplasty.

Isolated bronchial stenosis in infancy is rare, difficult to manage and becomes more complex when associated with congenital heart defects. Given the small luminal diameter and the proximity of the lung parenchyma to the bronchial lesion in infants, reconstruction of the bronchial stenosis is surgically challenging. We present 2 infant cases having isolated critical bronchial obstruction with congenital heart defects, which were successfully managed by primary one-stage slide bronchoplasty with concomitant heart repair. Both cases had excellent results after the surgery and did well at home without requiring respiratory support.

Absorbable Microplate Externally Suspending Bronchomalacia in Congenital Heart Disease Infant.

To evaluate the feasibility and efficacy of external suspension with absorbable poly-l-lactic acid material shaping microplates for infants with severe bronchomalacia and congenital heart disease. From November 2017 to January 2019, 11 continual patients with severe bronchomalacia and congenital heart disease underwent bronchial membrane external suspension together with cardiovascular surgery. An absorbable plate made with poly-l-lactic acid material was used as the shaping fixation material in all patients. Data included the details of the operation, and clinical results were collected. The mean age was 1.2 ± 1.0 years, and the mean weight was 7.7 ± 2.9 kg. The patients with cardiac malformations were operated on under low-temperature cardiopulmonary bypass (CPB) through median sternotomy. There were no in-hospital deaths. The CPB time, mechanical ventilation time, and length of intensive care unit stay were 123.9 ± 36.9 min, 20.7 ± 19.4 h, and 71.6 ± 54.9 h, respectively. Two patients underwent surgery through a left posterolateral incision without CPB. One was a double aortic arch repair, and the other was only bronchial membrane external suspension with prior IAA repair. No patients needed ECMO support. The mean follow-up time was 12.1 ± 5.6 months, and no patients were lost to follow-up. No cases of late death were noted, and no patients needed reoperation. According to the CT scans, no patients had bronchial restenosis. External bronchial membrane suspension with an absorbable poly-l-lactic acid material shaping plate, which had better histocompatibility, for infants with severe bronchomalacia and congenital heart disease was a safe and feasible procedure.

Combined use of Neurally Adjusted Ventilatory Assist (NAVA) and Vertical Expandable Prostethic Titanium Rib (VEPTR) in a patient with Spondylocostal dysostosis and associated bronchomalacia.

Jarcho-Levin syndrome is a rare disorder characterised by defects in vertebral and costal segmentation of varying severity. Respiratory complications are the main cause of death or severe comorbidity due to a restrictive rib cage. A 3 months old infant with Spondylocostal dysostosis and associated bronchomalacia experiencing severe asynchrony during the weaning process is reported. The Neurally Adjusted Ventilatory Assist (NAVA) mode was used to improve adaptation to mechanical ventilation after Vertical Expandable Prosthetic Titanium Ribs (VEPTRs) were implanted. The synchrony achieved with the NAVA mode allowed a decrease of the sedoanalgesia he received. A follow-up CT scan showed a reduction in the volume of the posterobasal atelectasis. The evolution of this patient suggests that the combined use of VEPTR for thoracic expansion and ventilation using NAVA can favour the global improvement. This mode could be an option to consider in selected patients with difficult weaning from mechanical ventilation in paediatric intensive care units.

Broncomalacia del adulto: entidad no tan infrecuente que mejora con presión positiva continua de la vía aérea / Bronchomalacia in adults: an infrequent entity that improves with continuous positive pressure on the airway

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Bronchoscopy findings in recurrent croup: A systematic review and meta-analysis.

IMPORTANCE: The etiology of recurrent croup is often anatomic. Currently there is no set criteria for determining who should undergo diagnostic bronchoscopy and which patients are at most risk for having a clinically significant finding. Few studies have addressed these questions. OBJECTIVE: To identify risk factors for clinically significant findings on bronchoscopy in children with recurrent croup and the frequency of bronchoscopy findings in general. DATA SOURCES: PUBMED, Ovid MEDLINE, EMBASE. STUDY SELECTION: Articles addressing bronchoscopy in children with recurrent croup, up to July 2016, were reviewed. Related keywords and medical subject headings were used during the search. The abstracts were reviewed to determine suitability for inclusion based on a set of criteria. Manual crosscheck of references was performed. DATA EXTRACTION: We analyzed the bronchoscopy findings of individual patients in each study and their associated risk factors when available. RESULTS: We reviewed 11 articles, published between 1992 and 2016, including 885 patients (654 males, 237 females). Only 5 studies, including 455 patients, had sufficient data for meta-analysis. Our study revealed that the three most common bronchoscopy findings were subglottic stenosis, reflux changes, and broncho/tracheomalacia. Only 8.7% of patients were noted to have clinically significant findings on bronchoscopy. Meta analysis showed an association between significant bronchoscopy findings and History of Intubation [OR = 5.17, 95% CI 2.65-10.09], Inpatient Consultation [OR = 4.01, 95% CI 1.44-11.20], Age < 3 [OR = 3.22, 95% 1.66-6.27], Age < 1 [OR = 2.86, 95% CI 1.28-6.40], and Prematurity [OR = 2.90, 95% CI 1.39-6.06]. Our study found a high incidence of a History of GERD (20%) and Asthma/Allergies (35%) among patients with recurrent croup, but these variables did not reach statistical significance in patients with significant bronchoscopy findings ([OR = 1.62, 95% CI 0.79-3.30], [OR = 0.57, 95% CI 0.30-1.08] respectively). CONCLUSION: The risk factors most associated with clinically significant bronchoscopy findings in recurrent croup are Intubation, Inpatient Consultation, Age < 3, Age <1, and Prematurity. A History of GERD and Asthma/Allergy, though highly prevalent in recurrent croup patients, were not statistically associated with significant bronchoscopy findings. RELEVANCE: The results should guide physicians in selecting which recurrent croup patients are most at risk for significant findings and thus may warrant bronchoscopy.

Condroma endobronquial: un caso atípico de obstrucción bronquial / Endobronchial chondroma: An unusual case of bronchial obstruction

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Airway Disease and Management in Bronchopulmonary Dysplasia.

This article presents an overview of the diagnosis and management of airway problems encountered in infants with severe bronchopulmonary dysplasia (BPD). Respiratory failure in premature infants develops as a result of parenchymal and airway diseases. The survival of increasingly premature infants and the ventilatory support required by premature lungs may result in airway disease. The management of respiratory failure depends on whether it is primarily caused by parenchymal versus airway diseases. Continuous airway pressure early in the neonatal period has favorably changed the incidence of BPD. This article discusses the indications, timing, and guidelines for care of tracheotomy.

Outcomes of slide tracheoplasty in 101 children: a 17-year single-center experience.

OBJECTIVE: Our study describes the results from surgical slide tracheoplasty (STP) in children with long segment tracheal stenosis. METHODS: Demographic and preoperative conditions, operative details, and outcome measures, including the need for endoscopic airway intervention and mortality, were collected for children undergoing STP between February 1995 and December 2012. RESULTS: One hundred one patients (median age, 5.8 months; range, 5 days-15 years) underwent STP. Seventy-two patients (71.3%) had associated cardiovascular anomalies. Preoperative ventilation was necessary in 56 patients (55.4%), whereas extracorporeal membrane oxygenation was required in 10 patients (9.9%). Abnormal bronchial arborization was present in 39 children (38.6%), which included 13 patients (12.8%) with an anomalous right upper lobe bronchus and 17 patients (16.8%) with tracheal trifurcation. Airway stenosis extended into 1 or both bronchi in 24 patients (23.7%) and preoperative malacia was present in 24 patients (23.7%). STP was extended into the bronchus in 47 patients (46.5%). Overall survival was 88.2% (mortality in 12 patients). Post-STP balloon dilation was necessary in 45 patients (44.6%) and stenting was required in 22 patients (21.8%). Multivariate analysis revealed preoperative extracorporeal membrane oxygenation (P < .05), preoperative malacia (P < .001), and bronchial stenosis (P < .05) to be adverse predictors of survival. Preoperative malacia was a significant risk factor for stenting (P < .05). CONCLUSIONS: STP is a versatile and reliable technique associated with low morbidity and mortality when compared with previous strategies for children with long segment tracheal stenosis. The presence of preoperative bronchomalacia is a significant risk factor for death and postoperative stenting.

Bronchomalacia in dogs with myxomatous mitral valve degeneration.

BACKGROUND: Cough in the geriatric small breed dog with myxomatous mitral valve degeneration (MMVD), a large left atrium, and absence of heart failure often is attributed to compression of the left mainstem bronchus by the left atrium. Studies investigating this syndrome are lacking in dogs. HYPOTHESIS: Airway collapse is independent of left atrial enlargement. ANIMALS: A total of 16 dogs presenting with chronic cough in the absence of congestive heart failure. Group 1 dogs (n = 10) had moderate-to-severe left atrial enlargement based on an echocardiographically calculated left atrial:aortic surface area [LA:Ao(a)] > 6. Group 2 dogs (n = 6) had no to mild left atrial enlargement [LA:Ao(a) ≤ 6]. METHODS: Dogs were prospectively evaluated. CBC, biochemistry, urinalysis, cervical and thoracic radiographs, fluoroscopy, echocardiography, and bronchoscopy were performed. Bronchoscopic abnormalities were compared between groups using Fisher's Exact Test. P < .05 was considered significant. RESULTS: Fluoroscopy identified airway collapse in both groups. Bronchoscopic evidence of airway collapse >50% was observed in multiple bronchi with no difference between groups. All dogs had inflammation on airway cytology with respiratory infection in 1 dog in group 2. Left atrial size was interpreted radiographically as enlarged in 9 of 10 group 1 dog and in 2 of 6 group 2 dogs. VHS was above normal in both groups of dogs regardless of echocardiographic evidence of cardiomegaly. CONCLUSIONS: Results failed to identify an association between left atrial enlargement and airway collapse in dogs with MMVD, but did suggest that airway inflammation is common in dogs with airway collapse.

Protracted bacterial bronchitis in young children: association with airway malacia.

OBJECTIVE: To examine associated findings and clinical outcome in young children with prolonged cough, wheeze, and/or noisy breathing in whom high colony counts of potentially pathogenic bacteria were cultured from bronchoalveolar lavage (BAL) during diagnostic flexible fiberoptic bronchoscopy. STUDY DESIGN: This was a retrospective review of all medical records of children from infancy to 60 months of age seen in our specialty clinic from 1999 to 2009 with protracted cough, wheeze, and/or noisy breathing in whom BAL found ≥ 10(4) colony forming units per milliliter of potentially pathogenic bacteria. Children with other major diagnoses were excluded. RESULTS: With quantitative culture from BAL, ≥ 10(4) colony forming units per milliliter of Streptococcus pneumoniae, Haemophilus influenza, or Moraxella catarrhalis, separately or in combination, were found in 70 children. Neutrophilia was present in 87% of BALs. Tracheomalacia, bronchomalacia, or both was present in 52 children (74%). Symptoms were eliminated with antibiotics in all 61 children with follow-up data. Relapse and subsequent successful re-treatment occurred in 43 children. CONCLUSIONS: High colony counts of potentially pathogenic bacteria associated with neutrophilia in the BAL identifies protracted bacterial bronchitis. The predominance of airway malacia in these patients suggests an etiologic role for those airway anomalies. The potential for chronic airway damage from protracted bacterial bronchitis warrants further investigation.

Lower airway anomalies in infants with laryngomalacia.

OBJECTIVE: To study the prevalence of associated airway anomalies in infants presenting with moderate to severe laryngomalacia. METHODS: Eighty three (83) symptomatic infants with recurrent respiratory symptoms including wheeze and cough diagnosed as moderate to severe laryngomalacia based on their clinical and direct laryngoscopic findings were subjected to fiberoptic bronchoscopy (FOB) during the period March 2007 to February 2009 in the Department of Pulmonology, Institute of Child Health and Hospital for Children, Chennai, India. Analysis of the clinical features, radiological findings and associated lower airway anomalies by FOB was done. RESULTS: Lower airway anomalies were demonstrated in 40 (48%) infants of the study population. Tracheomalacia was the most common lower airway anomaly 24 (29%) followed by bronchomalacia 8(10%) and tracheobronchomalacia 6 (7%). (4:1:1). CONCLUSION: Infants with moderate and severe laryngomalacia should be evaluated with flexible fibreoptic bronchoscopy to rule out associated lower airway lesions.

Recombinant human DNase in children with airway malacia and lower respiratory tract infection.

BACKGROUND: Children with airway malacia often have protracted courses of airway infections, because dynamic airway collapse during coughing results in impaired mucociliary clearance. The aim of this study was to determine the effect of the mucolytic drug recombinant human deoxyribonuclease (rhDNase) on the recovery of respiratory symptoms in children with airway malacia and lower respiratory tract infection (LRTI). METHODS: In a randomized double-blind controlled clinical trial, 40 children with airway malacia and LRTI were randomly assigned to receive either 2.5 mg nebulized rhDNase or placebo twice daily for 2 weeks. The primary endpoint was the change in the cough diary score (CDS) (scale 0-5) from baseline to the second week of treatment. Secondary endpoints were VAS symptom scores for cough, dyspnea, and difficulty in expectorating sputum, need for an antibiotic course, and lung function data (FVC, FEV(1), FEF(75), R(int(e))). RESULTS: There was no significant difference in the mean change in CDSs from baseline between the rhDNase group and the placebo group (mean difference for daytime 0.19 (95% CI -0.53 to 0.90); for nighttime 0.38 (95% CI -0.30 to 1.05). Proportions of patients requiring antibiotics, and the mean changes in symptom scores and lung function from baseline did not significantly differ between both groups. CONCLUSION: Treatment with 2 weeks of nebulized rhDNase does not enhance recovery or reduce the need for antibiotics in children with airway malacia and LRTI. (Controlled-trials.com number, ISRCTN85366144).