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Título

Obstructive sleep apnea as a manifestation of relapsing polychondritis: case report

Introdução

Relapsing polychondritis (RP) is a rare connective tissue disease in which recurrent bouts of inflammation involve the cartilage of the ears, nose, larynx, tracheobronchial tree and cardiovascular system. The cause of RP is still unknown. It is considered an immune-mediated disease with an annual incidence around of 3.5 cases per million.

Objetivo

We report a 22 year-old woman complaining of snoring and nocturnal stridor associated with occasional awakenings and daytime sleepiness in the last 05 months.

Métodos

The patient had a tracheal t-tube due to tracheal stenosis. Stridor and snoring started after her t-tube was changed to a shorter cannula. Oropharyngeal inspection did not show any findings suggesting the reason for her OSA: Friedman stage 2, with tonsil size grade 1 and Mallampati grade 1. Her body mass index was 18.5 kg/m², and her awake oximetry was 98%.

Resultados

Polysomnography showed an of AHI: 103.1 events per hour, associated with reduced sleep efficiency (44.3%) and a large wake after sleep onset (WASO) time: 230.5 min. A Drug Induced Sleep Endoscopy (DISE) was performed to identify the site of the obstruction. Respiratory flow was simultaneously recorded using a nasal mask attached to a pneumotachograph (see video acessing QRcode). The patient underwent a spirometry that revealed a fixed inspiratory and expiratory obstruction. A thoracic CT scan showed narrowing of the bronchial tree. During DISE, obstruction at the level of the glottis was observed. The velopharynx, oropharynx, tongue and epiglottis had a normal appearance. A fibrotic aspect of lower airway distal to the t-tube was observed.

Conclusões

Due to the impossibility of t-tube opening during sleep, CPAP was titrated to 8cmH2O. Obstructive events were abolished (residual AHI = 4.7 events per hour). After 30 days, the patient referred important quality of sleep improvement and resolution of daytime sleepiness.

Palavras-chave

Sleep obstructive apnea, relapsing polychondritis, CPAP

Área

Relato de Caso

Autores

Mariana Delgado Fernandes, Viviane Vieira Passini, Cassio Batista Lacerda, Thais Cano, Bruno Sergio Vasconcelos, Marcela Yanagimori, Camila Bae Uneda, Pedro Rodrigues Genta, Geraldo Lorenzi Filho