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Hirayama disease is a rare, lower cervical myelopathy affecting young adults. It is responsible for pure distal motor impairment of the upper limbs, with slow progressive development in the metameric territories of C7 to T1. It is thought to be caused by movements involved in flexing the neck. Neutral position magnetic resonance imaging (MRI) looks for abnormal cervical curvature, atrophy with flattening of the cervical spine, anterior cord hyperintensity and especially a lack of posterior apposition of the dural sac. If the condition is suspected, an MRI in flexion should be performed to show anterior displacement of the cord and dural sac, enlargement of the posterior epidural space, an increase in flattening of the cord and congestion of the epidural veins. These dynamic abnormalities tend to disappear after evolving for 10 years. We report two confirmed cases and a probable case of Hirayama disease and discuss its physiopathology.

Background Endotracheal tube cuff underinflation contributes to microaspiration of contaminated oropharyngeal content, source of pneumonia. Overinflation exposes to airway damage. Intermittent manual adjustment of the cuff pressure (P cuff ) may delay the detection of under- or overinflation. Devices for automated continuous adjustment of P cuff are promising but some are inconvenient, expensive or even harmful. This prospective randomized controlled study tested whether the Tracoe Smart Cuff Manager TM reduced the rate of patients undergoing ≥1 episode of underinflation (P cuff <20 cmH 2 O), as compared with routine manual P cuff adjustment. Secondary endpoints included comparisons of the rate of patients with ≥1 overinflation episode (P cuff >30 cmH 2 O), of the incidence of under- and overinflation episodes and of their magnitude. Methods Patients likely to receive invasive mechanical ventilation for >48 hours because of acute brain injury were randomly allocated to receive, during 48 hours, automated P cuff adjustment (combined with manual adjustment) or manual adjustment alone. In each group, P cuff was measured with a dedicated manual manometer, at least every 8 hours. Results Sixty patients were analyzed (28 patients with automated and 32 with manual P cuff adjustment) for 506 measurements of P cuff (237 and 269, respectively). As compared with manual adjustment, automated adjustment of P cuff was associated with 1) a lower rate of patients with ≥1 episode of underinflation (63% and 18%, respectively, p<0.001), 2) a lower incidence of underinflation episodes (15% vs. 2%;p<0.001), 3) a lower rate of manual adjustments (77% vs. 56%;p<0.001) and 4) manual adjustments of lower magnitude (5.9±4.5 vs. 3.6±4.7 cmH 2 O;p<0.001). For overinflation, there was no significant between-groups differences (p>0.99). Conclusions The adjunction of a continuous P cuff control with the Tracoe Smart Cuff Manager TM to routine manual intermittent adjustment reduced both the incidence of P cuff underinflation episodes and their magnitude without provoking overinflation.

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