PEDIATRICS Vol. 107 No. 6 June 2001, pp. 1343-1345

Successful Management of Tracheotomized Patients With Chronic Saliva Aspiration by Use of Constant Positive Airway Pressure

Jonathan D. Finder, MD^*, Robert Yellon, MD, and Martin Charron, MD^§

From the Departments of ^* Pediatrics,  Pediatric Otolaryngology, and ^§ Nuclear Medicine, Children's Hospital of Pittsburgh, Pittsburgh, Pennsylvania.

	ABSTRACT

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Objective.  Management of chronic aspiration of saliva is a challenge to clinicians. The purpose of this report is to review the clinical course of 3 patients with tracheotomy who we have followed for at least 1 year and who have received constant positive airway pressure (CPAP) as a primary treatment for ongoing aspiration of saliva.

Methods.  Retrospective chart review.

Results.  We present here 3 patients with chronic congestion and persistent hypoxemia in whom a diagnosis of chronic aspiration of saliva was established by use of radionuclide salivagram. Each of these children had tracheotomy for treatment of airway obstruction. In an attempt to decrease chronic aspiration of saliva, we instituted constant positive pressure via tracheotomy. Repeat radionuclide salivagram performed on CPAP demonstrated a marked decrease in saliva aspiration. All patients experienced improvement in clinical symptoms and required only rare subsequent hospitalizations for respiratory disease.

Conclusion.  We suggest, based on this case series, that CPAP administered via a tracheotomy is an acceptable means of managing chronic salivary aspiration and that it may decrease respiratory complications in such patients.  Key words:  constant positive airway pressure, aspiration, tracheotomy, saliva, salivagram.

In children with neurologic impairment affecting the brainstem, dysfunctional swallowing and chronic aspiration of secretions can lead to substantial respiratory morbidity and are often the cause of death. Such patients include those with cerebral palsy and those with isolated lesions of the brainstem, such as the Arnold-Chiari malformation. When children are recognized as having clinically significant aspiration with swallowing, gastrostomy tube feedings are often required. Feeding exclusively via gastrostomy can reduce the amount of aspiration of food from the oropharynx but does not stop individuals from aspirating either saliva or material refluxed from the stomach. Fundoplication or gastrojejunal feedings can decrease gastroesophageal reflux and, thus, aspiration of stomach contents but does not prevent aspiration of saliva.¹ Although the recognition of reflux-induced lung disease has been well-described in the literature,² there is little in the literature discussing the role of ongoing aspiration of saliva in the absence of reflux and oral feedings. We believe that chronic aspiration of saliva leads to bronchospasm, ventilation-perfusion mismatch, and hypoxemia. A state of constant inflammation induced by ongoing aspiration of saliva also seems to lead to poor tolerance of respiratory viral infections.³ Patients with chronic aspiration require frequent hospitalizations for respiratory illnesses. We present here 3 patients who had undergone tracheotomy for upper airway obstruction. Each had dramatic improvement in their respiratory status after institution of constant positive airway pressure (CPAP) via tracheotomy. Radionuclide salivagram was used to demonstrate chronic salivary aspiration, and subsequent repeat studies demonstrated resolution of the aspiration with the use of tracheal CPAP.

We propose here use of intratracheal CPAP as an effective therapy for such patients. It has been associated with minimal morbidity and an apparently decreased rate of hospitalization for respiratory causes.

	METHODS

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Protocol for Salivagram

Technetium-labeled sulfur colloid (1.0 mCi) in 0.3 mL of saline is placed under the patient's tongue.⁴ The head is kept facing anteriorly. Imaging is performed with a -camera equipped with a LEAP Collimator (Siemens, Iselin, NJ) with standard acquisition parameters (dynamic: 30 seconds/image for 120 frames; static: 300 seconds/exposure). The field of view includes the mouth and stomach. Images are acquired sequentially every 30 seconds for 1 hour. At the end of 1 hour, 300-second views of the chest are acquired in the posterior and lateral positions. The study is terminated earlier if no more activity is noted in the mouth.

Cases

Case 1 H.B. is a 1-year-old girl diagnosed with CHARGE syndrome. She underwent tracheotomy at birth for choanal atresia, severe laryngomalacia, and severe subglottic stenosis. She was also found to have gastroesophageal reflux. She was referred to the pulmonology service and admitted to the hospital at 4 months of age, when she had persistent hypoxemia, diffuse wheezes, and crackles on auscultation, with areas of atelectasis on chest radiograph. She required frequent suctioning of clear secretions from the tracheotomy. A radionuclide salivagram demonstrated aspiration, and CPAP via tracheotomy was started. The level of CPAP was titrated using the salivagram, and a level of 8 cm of water was found to be optimal in decreasing aspiration. In the 17 months since the institution of CPAP therapy, H.B. has become more stable from the respiratory standpoint. She has had elective hospitalization for correction of choanal stenosis, surgical ligation of patent ductus arteriosus, gastrostomy tube placement, and fundoplication. She is maintained on CPAP 24 hours a day and remains in room air. Her mother notes that when H.B. discontinues CPAP, she immediately has to begin to suction her airway. Repeated contrast swallowing studies have shown aspiration with thin consistencies of barium, and, thus, she is fed entirely via gastrostomy tube. In the 17 months that we have observed H.B. on CPAP, she has been hospitalized only once for respiratory reasons, and aside from that brief, 3-day period of hospitalization, has not required supplemental oxygen therapy.

Case 2 J.D. is a 23-year-old male with Sanfilippo's syndrome (mucopolysaccharidosis type IIIA). J.D. was diagnosed with Sanfilippo's syndrome at age 3 years, when he was found to be losing developmental milestones. He experienced choking and repeated aspiration in his early teens and had a gastrostomy tube placed at age 15 years. He has been exclusively gastrostomy tube fed since that time. He was seen in pulmonology clinic for repeated episodes of wheezing and hypoxemia and was treated for asthma. At age 20 years, he developed upper airway obstruction from a thickened and redundant epiglottis believed to be secondary to his mucopolysaccharidosis. A tracheotomy was performed. After tracheotomy, J.D. had persistent bronchospasm and mild hypoxemia. He required frequent suctioning. He was discharged from the intensive care unit to home and returned to pulmonology clinic a few weeks later. At the time of his first follow-up visit, his mother described a constant need to suction his upper airway and had to sit at his bedside throughout the night. He was intermittently hypoxemic and required supplemental oxygen. A radionuclide salivagram showed severe salivary aspiration. CPAP was instituted, with immediate cessation of his constant need of suctioning. After 17 months on CPAP, he requires only infrequent suctioning and no supplemental oxygen. Follow-up radionuclide salivagram demonstrated that no saliva entered his trachea. His respiratory status has been stable since institution of this therapy, with no hospitalizations, use of bronchodilators, or steroids.

Case 3 H.S. is an 8-month-old girl with laryngomalacia and an idiopathic primary swallowing disorder. A tracheotomy was performed at 6 weeks of life for severe laryngomalacia. After surgery, H.S. had profuse clear secretions suctioned frequently from her tracheotomy. After recovery from the surgery, oral feedings were restarted, and the patient developed diffuse wheezing, hyperinflation, and hypoxemia. Oral feeding was discontinued and all feedings were given via nasogastric tube and later gastrostomy. A salivagram was performed. It demonstrated that the entire bolus entered the trachea and, subsequently, was expelled through the tracheotomy tube. She was discharged from hospital on gastrostomy tube feedings but without CPAP. She returned to the hospital 6 weeks later, having had a persistent oxygen requirement, with respiratory distress and fever. She was discharged the following day on CPAP of 6 cm of water. Attempts at discontinuation of CPAP have resulted in an increase in oxygen requirement and greatly increased the need for suctioning of the trachea. In the 12 months since institution of CPAP, the patient has not required hospitalization for respiratory distress. She requires supplemental oxygen therapy to maintain hemoglobin saturation in the mid-90s. Subsequent salivagrams have demonstrated aspiration without CPAP but no aspiration when CPAP is used.

	DISCUSSION

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The majority of respiratory morbidity in patients with cerebral palsy and other disorders of swallowing is related to ongoing aspiration. Patients who have undergone tracheotomy are at increased risk for swallowing dysfunction.^5,6 Tracheotomy affects both the mechanical positioning of the larynx controlled by the extrinsic laryngeal musculature and the neurophysiologic regulation of the intrinsic laryngeal musculature. When the larynx is elevated by the extrinsic laryngeal musculature during swallowing, the epiglottis is displaced posteriorly and inferiorly by the base of the tongue. The epiglottis closes against the aryepiglottic folds, and ligamentous attachments pull the false vocal cords and aryepilottic folds superiorly and toward the midline to contact the epiglottis. Placement of a tracheotomy limits the laryngeal elevation needed for closure of the supraglottic larynx, thus, increasing the propensity for aspiration.^5-7

Aspiration is also prevented by the vocal cord adductor reflex, which is responsible for approximation of the true vocal cords. In canine models, chronic tracheotomy has been shown to increase the threshold for stimulation of the superior laryngeal nerve to evoke the adductor reflex as well as prolong the latency of the reflex. Thus, tracheotomy alters the threshold and the transneuronal conduction time for the laryngeal adductor reflex.^5,6 Loss of vocal cord adduction also results in a weakened cough.

Muz et al⁸ noted that patients with tracheotomies aspirate less when the tracheotomy is occluded. The combination, therefore, of a primary swallowing disorder plus a tracheotomy can lead to an exacerbation of chronic aspiration. Aspiration of saliva leads to chronic obstructive lower airway disease, poor tolerance of respiratory viral infections, and recurrent need for hospitalization.³

Chronic aspiration of saliva has been treated with anticholinergic therapies, salivary gland excision, salivary duct ligation, and laryngotracheal separation.^9-11 The surgical options involving the salivary glands are effective in decreasing saliva production and, therefore, aspiration, but increase the risk of dental caries because saliva is bacteriostatic. The anticholinergic medications (glycopyrrolate and atropine) frequently lose efficacy after a short time. Laryngotracheal separation is extreme because patients cannot speak, although it is, theoretically, reversible. Intratracheal CPAP has many advantages over these alternatives: it has no increased risk over tracheotomy alone, does not have medication side effects, and allows the patient to retain his or her voice. In addition, CPAP therapy also has the advantage of decreasing atelectasis, to which these patients are predisposed.

The radionuclide salivagram has been extremely valuable in our institution in identifying patients who are candidates for intratracheal CPAP. Heyman⁴ introduced the salivagram in 1989 in an attempt to increase the detection of aspiration of small volumes of liquids. The salivagram is the most sensitive technique to detect aspiration during swallowing.^4,8,912-15 The concentration of technetium used in a salivagram is ~100 times higher than what is used typically in a gastric emptying scan (330 µCi/mL vs 3 µCi/mL), and, thus, theoretically, allows for detection of much smaller amount of aspiration into the lungs. Muz et al,¹⁶ using a concentration of 2.5 mCi of technetium-99m in 20 mL of water (125 µCi/mL), also reported a high detection rate of aspirationthe study being positive in 24 of 33 patients. The radiation dose is a fraction of the that of a barium swallowing study (it is equivalent to that of a standard chest radiograph). At our institution the yield of positive salivagram studies is ~50% and has the highest positivity rate of the various studies used to document aspiration (milk scan, barium swallow, and salivagram). Although the utility of the salivagram has been well established in the literature, this test is underutilized by pediatric clinicians. We suggest that this test be used in patients with persistent respiratory symptoms despite appropriate therapy. Patients with tracheotomy are at particular risk of saliva aspiration because the tracheotomy removes 1 of the airway protective mechanisms. A salivagram should be considered in any patient with tracheotomy and significant lower airway disease. These patients would potentially benefit from CPAP. An additional benefit of the salivagram is that the examination can be followed the same day by a liquid meal and a milk scan to evaluate for gastroesophageal reflux and aspiration.

This report is limited by its low patient number, and we do not believe that this management strategy can be thought of as definitive. However, we were struck by the dramatic response each patient had to the therapy and by the apparent change in the rate of hospitalization of each. Because it is easier to demonstrate the effect on ongoing saliva aspiration of CPAP therapy, we believe that most of the improvement in our patients is attributable to decreased contamination of the lower airways with aspirated material. In contrast, we have no way of knowing the effect of long-term CPAP on (presumed) decreased atelectasis and improved ventilation-perfusion matching, which is more difficult to demonstrate, but also a clinically relevant response.

We have found that institution of tracheal CPAP in patients with tracheotomy and chronic aspiration of saliva has greatly improved their clinical status and has diminished their need for repeated hospitalization. The relatively minor cost of delivery of intratracheal CPAP relative to the cost of repeated hospitalizations for respiratory illnesses makes this an extremely cost-effective therapy. We have found that parents have been happy to put up with the inconvenience of having to transport their child with a CPAP machine and battery once they realize the long-term benefits of the therapy in keeping their child healthy and out of the hospital. These cases demonstrate that administration of this therapy can safely reduce the incidence of serious respiratory complications in patients with tracheotomy and chronic aspiration of saliva.

	FOOTNOTES

Received for publication Apr 18, 2000; accepted Oct 2, 2000.

Reprint requests to (J.D.F.) Children's Hospital of Pittsburgh, 3705 Fifth Ave, Pittsburgh, PA 15213. E-mail: finder{at}pitt.edu

	ABBREVIATIONS

CPAP, constant positive airway pressure.

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