RESULTS

INTRODUCTION

ABSTRACT IMAGING

REFERENCES

CASE PRESENTATION

Alternative methods of oxygen supplementation are used frequently in the neonatal intensive care unit (NICU) to avoid long-term endotracheal intubation which can result in acquired subglottic stenosis. Nasal continuous positive airway pressure (CPAP) provides an effective alternative and is used routinely at many pediatric institutions. Nasal CPAP provides a system of less resistance to oxygen delivery, allowing better alveolar patency. There are, however, anecdotal reports in the literature indicating that nasal CPAP support may also cause significant nasal complications in an obligate nasal breathing population. Loftus and colleagues described eight patients with nasal deformities attributed to nasal CPAP use, including but not limited to, nasal vestibular stenosis1.

Iatrogenic nasal vestibular stenosis results from disruption of the nasal vestibular lining with resultant proliferation of granulation and fibrous tissue2. In addition to occurring in association with nasal CPAP use, it has been described as a result of previous nasal surgical procedures2,3, nasotracheal intubation4, nasal packing2, excessive cauterization for epistaxis5, and birth trauma6. Congenital cases of nasal pyriform aperature stenosis, a different embryologic entity, has been described7,8; however, nasal vestibular stenosis is an acquired process

Nasal CPAP is gaining popularity as a preferred means of ventilatory support in the NICU, and the potential complications of this are starting to be seen more frequently. DeRowe and colleagues reported a case of a 6 week old neonate with bilateral nasal synechiae after respiratory assistance in the form of nasal CPAP catheters for 3 weeks, ultimately requiring endoscopic synechiolysis, bilateral stenting, and repeated dialations9. Similarly, Smith and colleagues describe two cases of vestibular stenosis following the use of nasal CPAP. One case described was in a 4 month-old ex-26 week premature infant who was found to have bilateral nasal vestibular stenosis obstructing 80% of the nasal airway and causing significant cosmetic deformity. They reported another case of bilateral vestibular stenosis causing 95% of nasal airway obstruction with significant cosmetic deformity as well. Both required surgical correction of the nasal defects10. These reports and this case report suggest that the use of nasal CPAP may potentially lead to significant nasal airway obstruction.

Objective: To report a case of bilateral nasal vestibular stenosis from nasal CPAP/cannulause in a neonate.

Study Design: Case report and review of literature.

Methods: A detailed clinical history, CT imaging, intraoperative and post-operative photographs is presented and the current literature is reviewed.

Results: A female, born at 29 weeks, with history of 10 days of nasal CPAP and 37 days of nasal cannula use was transferred to the NICU from an outside hospital for otolaryngologicevaluation of respiratory distress. Immediately after birth, nasal catheters were passed without difficulty through both nasal passages. At nearly 7 weeks of age, nasal endoscopy revealed bilateral near complete vestibular stenosis right at the tip where the nasal CPAP/cannula was positioned. CT scan of the facial bones demonstrated an anterior soft tissue band which occluded both nasal airways. Direct laryngoscopy and bronchoscopy were normal. Nasal repair was successfully performed endoscopically with lysis, application of mitomycin c, and nasal stenting with endotracheal tubes.

Conclusions: Bilateral vestibular stenosis can be a complication of nasal CPAP/cannulause, which may lead to chronic sinonasal obstruction. Such complication among neonates is not well documented in the literature to date, and it is likely underdiagnosed. Further studies are needed to elucidate incidence of acquired vesibular stenosis from nasal CPAP/cannula.

CONCLUSIONOnly a few cases of nasal vestibular stenosis from neonatal CPAP administration have been reported to date. We present a case of bilateral, nearly complete vestibular stenosis which resulted after only 10 days of nasal CPAP and 37 days of nasal cannula use. Our patient was successfully surgically repaired by endoscopic lysis, application of mitomycin c, and nasal stenting. As endoscopic evaluation of the nose is not a standard practice in the NICU setting, it is likely that this complication is underdiagnosed. This may have long-term implications for future sinonasal problems. Further studies are needed to determine the exact incidence.

We present a case of a 1046 gram female, born at 29 weeks gestation by vaginal delivery due to pre-term labor. Apgar scores were 9 at both 1 and 5 minutes. The mother was given adequate doses of steroids prior to delivery. After birth, nasal CPAP was placed and nasal catheters were placed through both nares without difficulty. No intubation was required. Nasal CPAP was removed after 10 days and placed in an oxygen tent prior to nasal cannula. The neonate was on nasal cannula for a total of 37 days prior to transfer to Columbus Children’s Hospital for evaluation of respiratory distress with feeding and failure to thrive.

Upon arrival at nearly 7 weeks of age, otolaryngologic consultation was obtained. On physical examination, the neonate was in no acute distress on 1/16th liter of oxygen by nasal cannula. Stertor but no stridor was present. A complete head and neck physical examination was performed. Multiple attempts were made to pass nasal suction catheters through both nares, however, resistance was met in the anterior nares. Flexible fiberopticnasal endoscopy at the bedside revealed that there was a wall of scar tissue between both the septum and inferior turbinate bilaterally. The scope was placed through a very small opening in the scar tissue which allowed the scope to be passed distally. The nasopharynx, oropharynx, larynx, and hypopharynx were all within normal limits.

Computed tomography (CT) scan of the facial bones was performed which demonstrated bilateral soft tissue densities in the anterior nasal cavity as shown in Figure 1. Airway fluroscopy and video swallow study were normal.

The patient was taken to the operating room where direct laryngoscopy and bronchoscopywere normal. Nasal endoscopy revealed bilateral scar bands causing near-complete nasal obstruction as shown in Figure 2. Endoscopic lysis was performed (Figure 3), mitomycin c was applied, and nasal stents using endotracheal tubes were placed. Two weeks later the stents were removed and mitomycin c was re-applied (Figure 4). Repeat nasal endoscopy one month post-operatively demonstrated patent nasal passages other than one small synechiae which was cleaved. Eight months post-operatively, the patient is asymptomatic and doing well.

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1. Loftus BC, Ahn J, Haddad J. Neonatal nasal deformities secondary to nasal continuous positive airway pressure. Laryngoscope 1994; 104: 1019-22.

2. Karen M, Chang E, Keen. Auricular composite grafting to repair nasal vestibular stenosis. Otolaryngol Head Neck Surg 2000; 122: 529-32.

3. Adamson PA, McGraw-Wall BL, Strecker HD, et al. Analysis of nasal or flow following repair of vestibular stenosis. J Otolaryngol 1998; 27 (4): 200-5.

4. Gowdar K, Bull MJ, Schreiner JA, Lemons EL et al. Nasal deformities in neonates. Their occurrence in those treated with nasal continuous positive airway pressure and nasal endotracheal tubes. Am J Dis Child 1980; 134: 954-57.

5. Bajaj PS, Bailey BN. Stenosis of the nostrils: a report of three cases. Br J Plast Surg 1969; 22: 269-73.6. Jablon JH, Hoffman JF. Birth trauma causing nasal vestibular stenosis. Arch Orolaryngol Head Neck Surg 1997;

123: 1004-6.7. Ramada HH, Ortiz O. Congenital nasal pyriform aperture (bony inlet) stenosis. Otolaryngol Head Neck Surg

1995; 113: 286-9.8. Van Den Abbeele T, Triglia JM, Francois M, et al. Congenital nasal pyriform aperture stenosis: diagnosis and

management of 20 cases. Ann Otol Rhinol Laryngol 2001; 110: 70-5.9. DeRowe A, Landsberg R, Fishman G, Halperin D., et al. Neonatal iatrogenic nasal obstruction. Int J Pediatr

Otorhinolaryngol 2004; 68: 613-17.10. Smith LP, Roy S. Treatment strategy for iatrogenic nasal vestibular stenosis in young children. Int J Pediatr

Otorhinolaryngol 2006; 70: 1369-73.

Left Nares POD #14Stents removed

Right nares POD #14Stents removed

Left nares after lysisRight nares after lysis

Left naresRight nares

FIGURE 1: Pre-Operative CT (axial and coronal) demonstrated soft tissue densities in both nasal passages.

FIGURE 2

FIGURE 3

FIGURE 4

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Bilateral vestibular stenosis from Bilateral vestibular stenosis from nasal CPAP and nasal CPAP and cannulacannula

Jatana, KR; Elmaraghy, CA. Department of Otolaryngology—Head and Neck Surgery

Columbus Children’s Hospital and The Ohio State University Medical Center, Columbus, Ohio, USA