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Spasmodic dysphonia: An overview of clinical features and treatment options

      Abstract

      Spasmodic dysphonia (SD) is considered a rare focal laryngeal dystonia characterized by task-specific voice dysfluency resulting from selective intrinsic laryngeal musculature hyperfunction. Symptoms may be attenuated by a sensory trick. Although SD can be seen at times in generalized dystonia syndrome, it is typically a sporadic phenomenon, and the involvement of the laryngeal adductor muscles is more common than that of the abductor muscles. This research reviews the literature for the pathogenesis, clinical characteristics, treatment options, and current management methods of SD. Technological advances have enabled clinicians to better understand the connection between laryngeal function and dysfunction. Refinements in imaging and genetic investigation techniques have helped better understand the underlying mechanisms of this neurolaryngology disorder. Currently, the standard of care for SD is the symptomatic management of botulinum toxin (BT) chemodenervation. This is supported by a large body of literature attesting to its efficacy in many different research studies, particularly in the uncomplicated adductor form of the disorder. Efforts towards surgical treatment predate the development of BT treatment by a decade, but the long-term efficacy has not been proven and, further research is expected. Symptom relief in patients with abductor SD and dystonia with tremors after surgical and BT treatments and those in patients remains suboptimal.

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      References

        • Mackenzie M.
        Hoarseness, loss of voice, and stridulous breathing, in relation to nervo-muscular affectations of the larynx.
        2nd ed. John Churchill & Sons, London1868
        • Cannito M.P.
        • Johnson J.P.
        Spastic dysphonia: a continuum disorder.
        J Commun Disord. 1981; 14: 215-233
        • Hyodo M.
        • Hisa Y.
        • Nishizawa N.
        • Omori K.
        • Shiromoto O.
        • Yumoto E.
        • et al.
        The prevalence and clinical features of spasmodic dysphonia: a review of epidemiological surveys conducted in Japan.
        Auris Nasus Larynx. 2021; 48: 179-184
        • Robe E.
        • Brumlik J.
        • Moore P.
        A study of spastic dysphonia. Neurologic and electroencephalographic abnormalities.
        Laryngoscope. 1960; 70: 219-245
        • Dedo H.H.
        Recurrent laryngeal nerve section for spastic dysphonia.
        Ann Otol Rhinol Laryngol. 1976; 85: 451-459
        • Bielamowicz S.
        • Ludlow C.L.
        Effects of botulinum toxin on pathophysiology in spasmodic dysphonia.
        Ann Otol Rhinol Laryngol. 2000; 109: 194-203
        • Ludlow C.L.
        • Naunton R.F.
        • Sedory S.E.
        • Schulz G.M.
        • Hallett M.
        Effects of botulinum toxin injections on speech in adductor spasmodic dysphonia.
        Neurology. 1988; 38: 1220-1225
        • Adler C.H.
        • Bansberg S.F.
        • Krein-Jones K.
        • Hentz J.G.
        Safety and efficacy of botulinum toxin type B (Myobloc) in adductor spasmodic dysphonia.
        Mov Disord. 2004; 19: 1075-1079
        • Blitzer A.
        Spasmodic dysphonia and botulinum toxin: experience from the largest treatment series.
        Eur J Neurol. 2010; 17: 28-30
        • Hyodo M.
        • Nagao A.
        • Asano K.
        • Sakaguchi M.
        • Mizoguchi K.
        • Omori K.
        • et al.
        Botulinum toxin injection into the intrinsic laryngeal muscles to treat spasmodic dysphonia: a multicenter, placebo-controlled, randomized, double-blinded, parallel-group comparison/open-label clinical trial.
        Eur J Neurol. 2021; 28: 1548-1556
        • Balint B.
        • Bhatia K.P.
        Dystonia: an update on phenomenology, classification, pathogenesis and treatment.
        Curr Opin Neurol. 2014; 27: 468-476
        • Ozelius L.J.
        • Hewett J.W.
        • Page C.E.
        • Bressman S.B.
        • Kramer P.L.
        • Shalish C.
        • et al.
        The early-onset torsion dystonia gene (DYT1) encodes an ATP-binding protein.
        Nat Genet. 1997; 17: 40-48
        • Klein C.
        • Brin M.F.
        • de Leon D.
        • Limborska S.A.
        • Ivanova-Smolenskaya I.A.
        • Bressman S.B.
        • et al.
        De novo mutations (GAG deletion) in the DYT1 gene in two non-Jewish patients with early-onset dystonia.
        Hum Mol Genet. 1998; 7: 1133-1136
        • Bressman S.B.
        Dystonia update.
        Clin Neuropharmacol. 2000; 23: 239-251
        • Walker R.H.
        • Shashidharan P.
        Developments in the molecular biology of DYT1 dystonia.
        Mov Disord. 2003; 18: 1102-1107
        • Blitzer A.
        • Brin M.F.
        • Simonyan K.
        • Ozelius L.J.
        • Frucht S.J.
        Phenomenology, genetics, and CNS network abnormalities in laryngeal dystonia: a 30-year experience.
        Laryngoscope. 2018; 128: S1-S9
        • Guiry S.
        • Worthley A.
        • Simonyan K.
        A separation of innate and learned vocal behaviors defines the symptomatology of spasmodic dysphonia.
        Laryngoscope. 2019; 129: 1627-1633
        • Kirke D.N.
        • Frucht S.J.
        • Simonyan K.
        Alcohol responsiveness in laryngeal dystonia: a survey study.
        J Neurol. 2015; 262: 1548-1556
        • Simonyan K.
        • Ludlow C.L.
        Abnormal activation of the primary somatosensory cortex in spasmodic dysphonia: an fMRI study.
        Cereb Cortex. 2010; 20: 2749-2759
        • Ali S.O.
        • Thomassen M.
        • Schulz G.M.
        • Hosey L.A.
        • Varga M.
        • Ludlow C.L.
        • et al.
        Alterations in CNS activity induced by botulinum toxin treatment in spasmodic dysphonia: an H215O PET study.
        J Speech Lang Hear Res. 2006; 49: 1127-1146
        • Haslinger B.
        • Erhard P.
        • Dresel C.
        • Castrop F.
        • Roettinger M.
        • Ceballos-Baumann A.O.
        Silent event-related" fMRI reveals reduced sensorimotor activation in laryngeal dystonia.
        Neurology. 2005; 65: 1562-1569
        • Battistella G.
        • Fuertinger S.
        • Fleysher L.
        • Ozelius L.J.
        • Simonyan K.
        Cortical sensorimotor alterations classify clinical phenotype and putative genotype of spasmodic dysphonia.
        Eur J Neurol. 2016; 23: 1517-1527
        • Blitzer A.
        • Brin M.F.
        • Stewart C.F.
        Botulinum toxin management of spasmodic dysphonia (laryngeal dystonia): a 12-year experience in more than 900 patients.
        Laryngoscope. 1998; 108: 1435-1441
        • Schweinfurth J.M.
        • Billante M.
        • Courey M.S.
        Risk factors and demographics in patients with spasmodic dysphonia.
        Laryngoscope. 2002; 112: 220-223
        • Kodama N.
        • Sanuki T.
        • Miyamoto T.
        • Tashiro J.
        Effects and differences of voice therapy on spasmodic dysphonia and muscle tension dysphonia: a retrospective pilot study.
        J Voice. 2021; S0892-1997: 00236-00238
        • Hyodo M.
        • Asano K.
        • Nagao A.
        • Hirose K.
        • Nakahira M.
        • Yanagida S.
        • et al.
        Botulinum toxin therapy: a series of clinical studies on patients with spasmodic dysphonia in Japan.
        Toxins. 2021; 13: 840https://doi.org/10.3390/toxins13120840
        • Biller H.F.
        • Som M.L.
        • Lawson W.
        Laryngeal nerve crush for spastic dysphonia.
        Ann Otol Rhinol Laryngol. 1979; 88: 531-532
        • Levine H.L.
        • Wood B.G.
        • Batza E.
        • Rusnov M.
        • Tucker H.M.
        Recurrent layngeal nerve section for spasmodic dysphonia.
        Ann Otol Rhinol Laryngol. 1979; 88: 527-530
        • Fritzell B.
        • Hammarberg B.
        • Schiratzki H.
        • Haglund S.
        • Knutsson E.
        • Martensson A.
        Long-term results of recurrent laryngeal nerve resection for adductor spasmodic dysphonia.
        J Voice. 1993; 7: 172-178
        • Aronson A.E.
        • De Santo L.W.
        Adductor spastic dysphonia: three years after recurrent laryngeal nerve resection.
        Laryngoscope. 1983; 93: 1-8
        • Dedo H.H.
        • Izdebski K.
        Evaluation and treatment of recurrent spasticity after recurrent laryngeal nerve section. A preliminary report.
        Ann Otol Rhinol Laryngol. 1984; 93: 343-345
        • Netterville J.L.
        • Stone R.E.
        • Rainey C.
        • Zealear D.L.
        • Ossoff R.H.
        Recurrent laryngeal nerve avulsion for treatment of spastic dysphonia.
        Ann Otol Rhinol Laryngol. 1991; 100: 10-14
        • Schiratzki H.
        • Fritzell B.
        Treatment of spasmodic dysphonia by means of resection of the recurrent laryngeal nerve.
        Acta Otolaryngol Suppl. 1988; 449: 115-117
        • Murry T.
        • Woodson G.E.
        Combined-modality treatment of adductor spasmodic dysphonia with botulinum toxin and voice therapy.
        J Voice. 1995; 9: 460-465
        • Blitzer A.
        • Brin M.F.
        • Fahn S.
        • Lange D.
        • Lovelace R.E.
        Botulinum toxin (BOTOX) for the treatment of "spastic dysphonia" as part of a trial of toxin injections for the treatment of other cranial dystonias.
        Laryngoscope. 1986; 96: 1300-1301
        • de Paiva A.
        • Meunier F.A.
        • Molgo J.
        • Aoki K.R.
        • Dolly J.O.
        Functional repair of motor endplates after botulinum neurotoxin type A poisoning: biphasic switch of synaptic activity between nerve sprouts and their parent terminals.
        Proc Natl Acad Sci U S A. 1999; 96: 3200-3205
        • Hallett M.
        How does botulinum toxin work?.
        Ann Neurol. 2000; 48: 7-8
        • Byrnes M.L.
        • Thickbroom G.W.
        • Wilson S.A.
        • Sacco P.
        • Shipman J.M.
        • Stell R.
        • et al.
        The corticomotor representation of upper limb muscles in writer's cramp and changes following botulinum toxin injection.
        Brain. 1998; 121: 977-988
        • Gilio F.
        • Curra A.
        • Lorenzano C.
        • Modugno N.
        • Manfredi M.
        • Berardelli A.
        Effects of botulinum toxin type A on intracortical inhibition in patients with dystonia.
        Ann Neurol. 2000; 48: 20-26
        • Hogikyan N.D.
        • Wodchis W.P.
        • Spak C.
        • Kileny P.R.
        Longitudinal effects of botulinum toxin injections on voice-related quality of life (V-RQOL) for patients with adductory spasmodic dysphonia.
        J Voice. 2001; 15: 576-586
        • Rubin A.D.
        • Wodchis W.P.
        • Spak C.
        • Kileny P.R.
        • Hogikyan N.D.
        Longitudinal effects of Botox injections on voice-related quality of life (V-RQOL) for patients with adductory spasmodic dysphonia: part II.
        Arch Otolaryngol Head Neck Surg. 2004; 130: 415-420
        • Bielamowicz S.
        • Stager S.V.
        • Badillo A.
        • Godlewski A.
        Unilateral versus bilateral injections of botulinum toxin in patients with adductor spasmodic dysphonia.
        J Voice. 2002; 16: 117-123
        • Dharia
        • Bielamowicz S.
        Unilateral versus bilateral botulinum toxin injections in adductor spasmodic dysphonia in a large cohort.
        Laryngoscope. 2020; 130: 2659-2662
        • Ludlow C.L.
        • Naunton R.F.
        • Fujita M.
        • Sedory S.E.
        Spasmodic dysphonia: botulinum toxin injection after recurrent nerve surgery.
        Otolaryngol Head Neck Surg. 1990; 102: 122-131
        • Sulica L.
        • Blitzer A.
        • Brin M.F.
        • Stewart C.F.
        Botulinum toxin management of adductor spasmodic dysphonia after failed recurrent laryngeal nerve section.
        Ann Otol Rhinol Laryngol. 2003; 112: 499-505
        • Berke G.S.
        • Blackwell K.E.
        • Gerratt B.R.
        • Verneil A.
        • Jackson K.S.
        • Sercarz J.A.
        Selective laryngeal adductor denervation-reinnervation: a new surgical treatment for adductor spasmodic dysphonia.
        Ann Otol Rhinol Laryngol. 1999; 108: 227-231
        • Berke G.S.
        • Blumin J.H.
        Spasmodic dysphonia: therapeutic options.
        Curr Opin Otolaryngol Head Neck Surg. 2000; 8: 509-513
        • Allegretto M.
        • Morrison M.
        • Rammage L.
        • Lau D.P.
        Selective denervation: reinnervation for the control of adductor spasmodic dysphonia.
        J Otolaryngol. 2003; 32: 185-189
        • Nakamura K.
        • Muta H.
        • Watanabe Y.
        • Mochizuki R.
        • Yoshida T.
        • Suzuki M.
        Surgical treatment for adductor spasmodic dysphonia–efficacy of bilateral thyroarytenoid myectomy under microlaryngoscopy.
        Acta Oto-laryngol. 2008; 128: 1348-1353
        • Tsuji D.H.
        • Chrispim F.S.
        • Imamura R.
        • Sennes L.U.
        • Hachiya A.
        Impact in vocal quality in partial myectomy and neurectomy endoscopic of thyroarytenoid muscle in patients with adductor spasmodic dysphonia.
        Braz J Otorhinolaryngol. 2006; 72: 261-266
        • Tsuji D.H.
        • Takahashi M.T.
        • Imamura R.
        • Hachiya A.
        • Sennes L.,U.
        Endoscopic laser thyroarytenoid myoneurectomy in patients with adductor spasmodic dysphonia: a pilot study on long-term outcome on voice quality.
        J Voice. 2012; 26 (e7-12): 666
        • Schuering J.H.C.
        • Heijnen B.J.
        • Sjögren E.V.
        • Langeveld A.P.M.
        Adductor spasmodic dysphonia: botulinum toxin a injections or laser thyroarytenoid myoneurectomy? A comparison from the patient perspective.
        Laryngoscope. 2020; 130: 741-746
        • Nomoto M.
        • Tokashiki R.
        • Hiramatsu H.
        • Konomi U.
        • Motohashi R.
        • Sakurai E.
        • et al.
        The comparison of thyroarytenoid muscle myectomy and type II thyroplasty for spasmodic dysphonia.
        J Voice. 2015; 29: 501-506
        • Isshiki N.
        • Tsuji D.H.
        • Yamamoto Y.
        • Iizuka Y.
        Midline lateralization thyroplasty for adductor spasmodic dysphonia.
        Ann Otol Rhinol Laryngol. 2000; 109: 187-193
        • Isshiki N.
        • Haji T.
        • Yamamoto Y.
        • Mahieu H.F.
        Thyroplasty for adductor spasmodic dysphonia: further experiences.
        Laryngoscope. 2001; 111: 615-621
        • Isshiki N.
        • Yamamoto I.
        • Fukagai S.
        Type 2 thyroplasty for spasmodic dysphonia: fixation using a titanium bridge.
        Acta Oto-laryngol. 2004; 124: 309-312
        • Sanuki T.
        • Nishimura T.
        • Aburada T.
        • Fukushima M.
        Titanium bridge modification for type 2 thyroplasty to improve device mechanical & safety performance.
        Auris Nasus Larynx. 2021; S0385-8146: 00252
        • Sanuki T.
        • Yumoto E.
        Long-term evaluation of type 2 thyroplasty with titanium bridges for adductor spasmodic dysphonia.
        Otolaryngol Head Neck Surg. 2017; 157: 80-84
        • Shaw G.Y.
        • Sechtem P.R.
        • Rideout B.
        Posterior cricoarytenoid myoplasty with medialization thyroplasty in the management of refractory abductor spasmodic dysphonia.
        Ann Otol Rhinol Laryngol. 2003; 112: 303-306