Sleep-related stridor due to dystonic vocal cord motion and neurogenic tachypnea/tachycardia in multiple system atrophy
Identifieur interne : 004050 ( Main/Merge ); précédent : 004049; suivant : 004051Sleep-related stridor due to dystonic vocal cord motion and neurogenic tachypnea/tachycardia in multiple system atrophy
Auteurs : Roberto Vetrugno [Italie] ; Rocco Liguori [Italie] ; Pietro Cortelli [Italie] ; Giuseppe Plazzi [Italie] ; Claudio Vicini [Italie] ; Aldo Campanini [Italie] ; Roberto D'Angelo [Italie] ; Federica Provini [Italie] ; Pasquale Montagna [Italie]Source :
- Movement disorders [ 0885-3185 ] ; 2007.
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Abstract
Sleep-disordered breathing and sleep-related motor phenomena are part of the clinical spectrum of multiple system atrophy (MSA). Stridor has been attributed to denervation of laryngeal muscles or instead to dystonic vocal cord motion. We studied 3 patients with nocturnal stridor in the setting of MSA. All patients underwent nocturnal videopolysomnography (VPSG) with breathing and heart rate, O2 saturation and intra-esophageal pressure recordings, and simultaneous EMG recordings of the posterior cricoarytenoid, cricothyroid, and thyroarytenoid muscles and continuous vocal cord motion evaluation by means of fiberoptic laryngoscopy. VPSG/EMG and fiberoptic laryngoscopy documented normal vocal cord motion without denervation during wake and stridor only during sleep when hyperactivation of vocal cords adductors appeared in the absence of significant O2 desaturation. All patients had tachycardia and tachypnea and paradoxical breathing during sleep, erratic intercostalis and diaphragmatic EMG activity and Rem sleep behavior disorder. One of the patients had restless legs syndrome with periodic limb movement during sleep and excessive fragmentary hypnic myoclonus. In conclusion, our patients with MSA had nocturnal stridor due to sleep-related laryngeal dystonia. Stridor was associated with other abnormal sleep-related respiratory and motor disorders, suggesting an impairment of homeostatic brainstem integration in MSA.
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Morgagni-L. Pierantoni Hospital</s1><s2>Forlì</s2><s3>ITA</s3><sZ>5 aut.</sZ><sZ>6 aut.</sZ></inist:fA14><country>Italie</country><wicri:noRegion>Forlì</wicri:noRegion></affiliation></author><author><name sortKey="Campanini, Aldo" sort="Campanini, Aldo" uniqKey="Campanini A" first="Aldo" last="Campanini">Aldo Campanini</name><affiliation wicri:level="1"><inist:fA14 i1="02"><s1>ENT and Head and Neck Surgery Unit, G.B. Morgagni-L. Pierantoni Hospital</s1><s2>Forlì</s2><s3>ITA</s3><sZ>5 aut.</sZ><sZ>6 aut.</sZ></inist:fA14><country>Italie</country><wicri:noRegion>Forlì</wicri:noRegion></affiliation></author><author><name sortKey="D Angelo, Roberto" sort="D Angelo, Roberto" uniqKey="D Angelo R" first="Roberto" last="D'Angelo">Roberto D'Angelo</name><affiliation wicri:level="1"><inist:fA14 i1="01"><s1>Department of Neurological Sciences, University of Bologna</s1><s2>Bologna</s2><s3>ITA</s3><sZ>1 aut.</sZ><sZ>2 aut.</sZ><sZ>3 aut.</sZ><sZ>4 aut.</sZ><sZ>7 aut.</sZ><sZ>8 aut.</sZ><sZ>9 aut.</sZ></inist:fA14><country>Italie</country><wicri:noRegion>Bologna</wicri:noRegion></affiliation></author><author><name sortKey="Provini, Federica" sort="Provini, Federica" uniqKey="Provini F" first="Federica" last="Provini">Federica Provini</name><affiliation wicri:level="1"><inist:fA14 i1="01"><s1>Department of Neurological Sciences, University of Bologna</s1><s2>Bologna</s2><s3>ITA</s3><sZ>1 aut.</sZ><sZ>2 aut.</sZ><sZ>3 aut.</sZ><sZ>4 aut.</sZ><sZ>7 aut.</sZ><sZ>8 aut.</sZ><sZ>9 aut.</sZ></inist:fA14><country>Italie</country><wicri:noRegion>Bologna</wicri:noRegion></affiliation></author><author><name sortKey="Montagna, Pasquale" sort="Montagna, Pasquale" uniqKey="Montagna P" first="Pasquale" last="Montagna">Pasquale Montagna</name><affiliation wicri:level="1"><inist:fA14 i1="01"><s1>Department of Neurological Sciences, University of Bologna</s1><s2>Bologna</s2><s3>ITA</s3><sZ>1 aut.</sZ><sZ>2 aut.</sZ><sZ>3 aut.</sZ><sZ>4 aut.</sZ><sZ>7 aut.</sZ><sZ>8 aut.</sZ><sZ>9 aut.</sZ></inist:fA14><country>Italie</country><wicri:noRegion>Bologna</wicri:noRegion></affiliation></author></analytic><series><title level="j" type="main">Movement disorders</title><title level="j" type="abbreviated">Mov. disord.</title><idno type="ISSN">0885-3185</idno><imprint><date when="2007">2007</date></imprint></series></biblStruct></sourceDesc><seriesStmt><title level="j" type="main">Movement disorders</title><title level="j" type="abbreviated">Mov. disord.</title><idno type="ISSN">0885-3185</idno></seriesStmt></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Dystonia</term><term>Electromyography</term><term>Multiple system atrophy</term><term>Nervous system diseases</term><term>Polysomnography</term><term>Sleep</term><term>Stridor</term><term>Tachycardia</term><term>Vocal cord</term></keywords><keywords scheme="Pascal" xml:lang="fr"><term>Système nerveux pathologie</term><term>Atrophie multisystématisée</term><term>Dystonie</term><term>Sommeil</term><term>Corde vocale</term><term>Tachycardie</term><term>Polysomnographie</term><term>Electromyographie</term><term>Stridor</term></keywords></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Sleep-disordered breathing and sleep-related motor phenomena are part of the clinical spectrum of multiple system atrophy (MSA). Stridor has been attributed to denervation of laryngeal muscles or instead to dystonic vocal cord motion. We studied 3 patients with nocturnal stridor in the setting of MSA. All patients underwent nocturnal videopolysomnography (VPSG) with breathing and heart rate, O<sub>2</sub> saturation and intra-esophageal pressure recordings, and simultaneous EMG recordings of the posterior cricoarytenoid, cricothyroid, and thyroarytenoid muscles and continuous vocal cord motion evaluation by means of fiberoptic laryngoscopy. VPSG/EMG and fiberoptic laryngoscopy documented normal vocal cord motion without denervation during wake and stridor only during sleep when hyperactivation of vocal cords adductors appeared in the absence of significant O<sub>2</sub> desaturation. All patients had tachycardia and tachypnea and paradoxical breathing during sleep, erratic intercostalis and diaphragmatic EMG activity and Rem sleep behavior disorder. One of the patients had restless legs syndrome with periodic limb movement during sleep and excessive fragmentary hypnic myoclonus. In conclusion, our patients with MSA had nocturnal stridor due to sleep-related laryngeal dystonia. Stridor was associated with other abnormal sleep-related respiratory and motor disorders, suggesting an impairment of homeostatic brainstem integration in MSA.</div></front></TEI><affiliations><list><country><li>Italie</li></country></list><tree><country name="Italie"><noRegion><name sortKey="Vetrugno, Roberto" sort="Vetrugno, Roberto" uniqKey="Vetrugno R" first="Roberto" last="Vetrugno">Roberto Vetrugno</name></noRegion><name sortKey="Campanini, Aldo" sort="Campanini, Aldo" uniqKey="Campanini A" first="Aldo" last="Campanini">Aldo Campanini</name><name sortKey="Cortelli, Pietro" sort="Cortelli, Pietro" uniqKey="Cortelli P" first="Pietro" last="Cortelli">Pietro Cortelli</name><name sortKey="D Angelo, Roberto" sort="D Angelo, Roberto" uniqKey="D Angelo R" first="Roberto" last="D'Angelo">Roberto D'Angelo</name><name sortKey="Liguori, Rocco" sort="Liguori, Rocco" uniqKey="Liguori R" first="Rocco" last="Liguori">Rocco Liguori</name><name sortKey="Montagna, Pasquale" sort="Montagna, Pasquale" uniqKey="Montagna P" first="Pasquale" last="Montagna">Pasquale Montagna</name><name sortKey="Plazzi, Giuseppe" sort="Plazzi, Giuseppe" uniqKey="Plazzi G" first="Giuseppe" last="Plazzi">Giuseppe Plazzi</name><name sortKey="Provini, Federica" sort="Provini, Federica" uniqKey="Provini F" first="Federica" last="Provini">Federica Provini</name><name sortKey="Vicini, Claudio" sort="Vicini, Claudio" uniqKey="Vicini C" first="Claudio" last="Vicini">Claudio Vicini</name></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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