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An Analysis of Children with Tracheomalacia Treated With Ipratropium Bromide (Atrovent)

Thomas Gallagher, DO1; Steve Maturo, MD1; Shannon Fracchia, MD2; Christopher Hartnick, MD1

1Massachusetts Eye and Ear Infirmary, Harvard Medical School 2Massachusetts General Hospital, Harvard Medical School

INTRODUCTION DISCUSSIONRESULTSABSTRACT

Objective: Evaluate the treatment results of ipratropium bromide in children with tracheomalacia.

Setting: Tertiary care multidisciplinary aero digestive center

Design: Retrospective chart review of multidisciplinary aero digestive database.

Results: Complete data was available for 52 children diagnosed with tracheomalacia and treated with ipratropium bromide after having been referred for specialist care for symptoms that were severe enough to warrant bronchoscopy or for symptoms that were refractory to prior medical therapy. Mild tracheomalacia was diagnosed in 34 (65.3%) children while moderate tracheomalacia was seen in 18 (34.7%). Overall 32 (61.5%) children had improvement in their symptoms following treatment with ipratropium bromide.

Conclusion: In our retrospective review, symptoms attributed to mild/moderate tracheomalacia show improvement when treated with ipratropium bromide. Although the clinical symptoms of tracheomalacia overlap with many common pediatric aero digestive diagnoses, we have found that a thorough multidisciplinary approach with appropriate medical treatment can help most children alleviate their symptoms. Further controlled studies are warranted in order to optimize medical treatment strategies for children with tracheomalacia.

A retrospective chart review identified 52 children who had been diagnosed with tracheomalacia and treated with ipratropium. There were 22 girls and 30 boys. The average age was 4.2 years (range 6 months to 13 years.) The most common presenting symptom was cough (28), followed by recurrent croup (19), and stridor (4). Of those children with pre-existing diagnoses the most common were extra-esophageal reflux disease (16), asthma refractory to standard treatment (14), and recurrent croup (3).

Mild tracheomalacia was diagnosed in 34 (65.3%) children while moderate tracheomalacia was seen in 18 (34.7%). 28 out of 52 children had BAL fluid examined. In 17 children at least one pathogen was cultured. The most common pathogen was Moraxella catarrhalis (41%) followed by Streptococcus pneumoniae (18%). In 24 children lipen laden macrophage index (LLMI) was determined in the BAL fluid. Mean LLMI was 26.5 (range 0-120).

Overall 32 (61.5%) children had improvement in their symptoms following treatment with ipratropium bromide with 20 (58.8%) in the mild group showing improvement and 12 (66.6%) in the moderate group showing improvement. In children with a pre-existing diagnosis of reflux only 38% (6/16) improved with ipratropium treatment. There was no association with improvement in regards to BAL culture or LLMI.

This report describes our initial experience treating children with mild to moderate tracheomalacia with ipratropium bromide. A significant amount of children in our cohort had improved symptoms while taking this medication. Due to our initial findings we continue to administer ipratropium bromide to patients with mild to moderate tracheomalacia that have not had relief with previous therapy. It is unclear why ipratropium works in children with tracheomalacia. It is unknown whether ipratropium acts directly on the tracheal smooth muscle or if it provides improvement in lower airway dynamics allowing for symptom resolution.

There are obvious limitations to this study thus our conclusions must be tempered and followed with a prospective treatment analysis. The diagnosis of tracheomalacia is inherently subjective and no true grading criteria are universally accepted.7 A second limitation is the inherent weakness found with retrospective chart reviews. The analysis of chief complaints, previous diagnoses and treatments was thoroughly evaluated via a chart review, but we realize that the data may not be entirely complete. Finally, the administration of ipratropium and the duration of treatment were not controlled for thus limiting a true medication affect analysis.

Historically tracheomalacia has been treated with a “wait and see” approach where it is assumed that the majority of children will outgrow their symptoms. Many children though continue to have chronic symptoms and current medical treatment options are limited. Ipratropium may provide an effective medical treatment option.

This review received institutional review board approval. The study cohort included all children under the age of 18 who were diagnosed with tracheomalacia via rigid and flexible bronchoscopy and treated with ipratropium bromide between 2005 and 2009.

Bronchoscopy EvaluationRigid laryngoscopy/tracheoscopy was carried out using a 4mm rigid endoscope while flexible bronchoscopy was done through an LMA. Appropriate photos and videos were taken and stored during each individual endoscopy.

Tracheomalacia was defined as mild, moderate, and severe. Mild malacia was defined as less than 50 percent anterior tracheal wall collapse while moderate malacia was greater than 50 percent collapse. Severe malacia was defined as the anterior wall collapsing onto the posterior membranous segment of the trachea. The severity was determined by the attending pulmonologist and otolaryngologist at the time of the procedure.

Atrovent AdministrationIpratropium bromide (Atrovent) is an anti-cholinergic most commonly used to treat asthma in children. Children in our clinic are administered ipratropium either via nebulization or inhaler depending upon their age. Children under the age of 2 are given 125-250mcg nebulization treatment three times a day while children over the age of 2 are given 250-500mcg three times per day. Children under the age of 12 who can use a metered dose inhaler with spacer take 1 puff (17mcg per actuation) three times a day; those over 12 take 2 puffs three times a day. Each child underwent at least a 6 month trial of therapy prior to assessing effectiveness.

Our preliminary review of children with mild to moderate tracheomalacia shows modest results in symptom resolution in children with mild to moderate tracheomalacia. Further controlled studies will be necessary to determine the true effectiveness of ipratropium. Children with continuing refractory asthma and airway symptoms attributed to extra-esophageal reflux that are not improving with a medical regimen should be evaluated in a multi-disciplinary manner for tracheomalacia.

Tracheomalacia is weakness of the tracheal cartilage causing collapse of the anterior tracheal wall. Tracheomalacia is the most common congenital tracheal anomaly.1 The incidence of tracheomalacia is estimated to be 1 in 1500-2000 children.2-5

Tracheomalacia symptoms include cough, wheeze, recurrent lower airway infections, exercise intolerance, and respiratory insufficiency.6 The majority of children outgrow tracheomalacia by 2 years of age, but a significant proportion go undiagnosed for years and fail to outgrow their symptoms entirely.

Anecdotal experience in our clinic of children with tracheomalacia taking ipratropium bromide (Atrovent, Boehringer Ingelheim Pharmaceuticals, Germany) revealed improvement in symptoms. The purpose of this report is to describe the symptoms and operative findings in children diagnosed with tracheomalacia and treated with ipratropium.

METHODS AND MATERIALS

1. Carden KA, Boiselle PM, Waltz DA et al. Tracheomalacia and tracheobronchomalacia in children and adults: an in-depth review. Chest. 2005;127:984-1005.

2. Boogaard R, Huijsmans SH, Pilneneburg MWH et al. Tracheomalacia and bronchomalacia in children: incidence and patient characteristics.

3. Callahan CW. Primary tracheomalacia and gastroesophageal reflux in infants with cough. Clin Pediatr. 1998;37:725-31.

4. Masters IB, Change AB, Patterson L et al. Series of laryngomalacia, tracheomalacia, and bronchomalacia disorders and their associations with other conditions in children. Pediatric Pulmonol. 2002;34:189-95.

5. Finder JD. Primary bronchomalacia in infants and children. J Pediatr. 1997l130:59-66.

6. Weinberger M, Abu-Hasan M. Pseudo-asthma: when cough, wheezing, and dyspnea are not asthma. Pediatrics. 2007;120:855-64.

7. Mair EA, Parsons DS. Pediatric tracheobronchomalacia and major airway collapse. Ann Otol Rhinol Laryngol. 1992;101:300-309.

CONCLUSIONS

REFERENCES

Thomas Gallagher, DOMassachusetts Eye and Ear InfirmaryThomas_Gallagher @meei.harvard.eduPhone: 617 573 4206

CONTACT

Rigid endoscopic view reveals mild tracheomalacia.

Rigid endoscopic view reveals moderate tracheomalacia.