Simultaneous sleep study and nasoendoscopic investigation in a patient with obstructive sleep apnoea syndrome refractory to continuous positive airway pressure: a case report

Abstract

Introduction: The standard treatment for obstructive sleep apnoea syndrome is nasal continuous positive airway pressure. In most cases the obstruction is located at the oropharyngeal level, and nasal continuous positive airway pressure is usually effective. In cases of non-response to nasal continuous positive airway pressure other treatments like mandibular advancement devices or upper airway surgery (especially bi-maxillary advancement) may also be considered.

Case presentation: We report the case of a 38-year-old Caucasian man with severe obstructive sleep apnoea syndrome, initially refractory to nasal continuous positive airway pressure (and subsequently also to a mandibular advancement devices), in which the visualization of the upper airway with sleep endoscopy and the concomitant titration of positive pressure were useful in the investigation and resolution of sleep disordered breathing. In fact, there was a marked reduction in the size of his nasopharynx, and a paresis of his left aryepiglotic fold with hypertrophy of the right aryepiglotic fold. The application of bi-level positive airway pressure and an oral interface successfully managed his obstructive sleep apnoea.

Conclusion: This is a rare case of obstructive sleep apnoea syndrome refractory to treatment with nocturnal ventilatory support. Visualization of the endoscopic changes, during sleep and under positive pressure, was of great value to understanding the mechanisms of refractoriness. It also oriented the therapeutic option. Refractoriness to obstructive sleep apnoea therapy with continuous positive airway pressure is rare, and each case should be approached individually.

Figure 1

An example of a mandibular advancement device.

Figure 2

A cardiorespiratory study in the use of a mandibular advancement device. An evaluation at 4 months with a cardiorespiratory study in the use of a mandibular advancement device (first 3.5 h of study) showed no significant improvement in the patient's obstructive sleep apnoea syndrome (apnoea and hypopnoea index at 61.4/h and desaturation index of 42.1/h). A severe condition of obstructive sleep apnoea syndrome was observed with and without the use of the device.

Figure 3

Split-night polysomnography: Obstructive sleep apnoea syndrome refractoriness with continuous positive airway pressure and bilevel positive airway pressure, nasal mask. (Evaluation at 4 months). The first part of the night confirmed the severity of obstructive sleep apnoea syndrome (apnoea and hypopnoea index of 64.9/h with minimum O2 saturation of 29%). The second part allowed a gradual increase in positive pressure, first in continuous positive airway pressure for up to 16 cmH2O, then in bilevel positive airway pressure with a maximum inspiratory pressure of 24 cmH2O and a maximum expiratory pressure of 20 cmH2O. There were persistent obstructive events with marked desaturation (minimum O2 saturation of 45% in continuous mode and of 82% in bilevel mode).

Figure 4

A home nocturnal oximetry (in bilevel mode) with nasal mask. (Evaluation at 5 months). A home nocturnal oximetry (in bilevel mode, inspiratory pressure of 20 cmH2O and expiratory pressure of 12 cmH2O, and a gel face mask (Mojo) shows episodes of desaturation suggesting a large number of residual apnoea and/or hypopnoea events.

Figure 5

A facial computed tomography at 5.5 months following the initial presentation shows a smaller upper airway.

Figure 6

A facial computed tomography reformation at 5.5 months after the initial presentation shows an angular dysmorphia at the hypopharynx level.

Figure 7

(A) Sleep endoscopy before ventilation. Extended periods of vibration of the walls of the oropharynx related to snores were observed. With the establishment of positive pressure ventilation, the nasopharynx subocclusion persisted up to 24 cmH2O inspiratory pressure and 16 cmH2O expiratory pressure. An unrolling of the epiglottis that collapsed the airway and provoked periods of O2 desaturation was also noted. (C) Sleep nasoendoscopy under continuous positive airway pressure with P > 16 cmH2O at 6 months after the initial presentation. In this evaluation, a marked reduction of the size of the nasopharynx, and a paresis of the left aryepiglotic fold with hypertrophy of the right one were noted.

Figure 8

A polysomnography study during sleep endoscopy at 6 months after the initial presentation. A polysomnography and titration of ventilatory support pressures were also performed during sleep endoscopy.

Figure 9

A nocturnal oximetry under inspiratory pressure of 20 cmH20, expiratory pressure of 13 cmH20 with Oracle^® at 6.5 months after the initial presentation. A home oximetry under bilevel mode, inspiratory pressure of 20 cmH20 and expiratory pressure of 13 cmH20 and Oracle^® mask revealed a significant improvement in nocturnal desaturation episodes.