Case Presentation:
A previously healthy 2 year-old male was transported from home to our hospital following an unwitnessed powdered cinnamon aspiration. He was described as mottled and wheezing with visibly increased work of breathing. He was hypoxemic (SpO2 55%) in ambient air, necessitating escalation to non-invasive bi-level positive pressure ventilation and continuous albuterol nebulization. His first blood gas analysis, from a venous sample, found significant respiratory acidosis (pH 6.98, pCO2 112 mm Hg). After increasing respiratory support, an arterial sample revealed persistent respiratory acidosis (pH 7.19, pCO2 65.2 mm Hg) without significant metabolic contribution (bicarbonate 26 mmol/L, anion gap 14 mmol/L, lactate 1.26 mmol/L) and impaired oxygenation (pO2 66.2 mm Hg on 100% FiO2). Upon transfer to the PICU, he underwent endotracheal intubation and initiation of synchronized intermittent mandatory ventilation. His chest radiograph revealed lung hyper-expansion and bilateral patchy opacities sparing the apices (Figure 1A), and his ventilator scalars were consistent with increased airway resistance and lower airways obstruction. Intravenous methylprednisolone (loading dose of 2 mg/kg followed by 4 mg/kg/d) was initiated. By hospital day (HD) 4, his ventilator scalars demonstrated resolution of lower airways obstruction, and his hypercarbia improved, but his oxygenation index (OI) rose to the mid-20’s (Figure 1D). We transitioned him to airway pressure release ventilation (APRV) and started inhaled nitric oxide.
The first diagnostic flexible bronchoscopy with lavage (30 mL of normal saline instilled in total) was performed on HD 6. The airways of the left lung appeared more heavily deposited than the right, with moderate amounts of brown debris in all of the left lobes. The mucosa appeared normal, but with mild friability after the lavage. Analysis of the lavage fluid found increased white blood cells with a neutrophilic predominance and the moderate presence of lipid-laden macrophages; no analysis could be performed on the debris.
A chest radiograph attained just prior the bronchoscopy was later interpreted as showing pneumomediastinum and subcutaneous emphysema. Given worsening oxygenation (OI 32), the presence of air leak, ongoing intermittent obstructions of the endotracheal tube with mucoid cinnamon, and the need for additional bronchoalveolar lavages, the patient was deployed at bedside onto veno-venous extracorporeal membrane oxygenation (VV-ECMO, Figure 1B and 1D). On HD 7, he developed a new pneumothorax requiring chest tube placement.
He underwent repeated bronchoscopies on HD 9 and HD 10 with the goal of debris removal through high volume, total left lung lavage (120-150 mL normal saline instilled in total per procedure). At that time, the left lung demonstrated significantly worse mucosal inflammation than on HD 6, with a large quantity of visible debris, most heavily deposited in the left lower lobe (Figure 1E). After each lavage, we instilled surfactant (1.25ml/kg and 0.625ml/kg on HD 9 and HD 10, respectively). Each bronchoscopy was accompanied by an immediate worsening in oxygenation followed by slow, steady improvement; this was most dramatic on HD 10, with increasing lung compliance and tolerance of extracorporeal oxygen blender weaning. The patient was decannulated on HD 14, extubated on HD 18, and transferred on HD 28 to a sub-acute inpatient rehabilitation program, requiring no supplemental respiratory support. He completed the program and is now at home. At a pulmonary medicine follow-up visit 6 months after the aspiration, his chest radiograph demonstrated persistence of patchy opacities in the right lung base, with no abnormalities on the left (Figure 1C), which had directly received the bronchoalveolar lavages and surfactant. He was doing well, with no ongoing respiratory symptoms or need for respiratory support or medications.