Spread of segmental/multifocal idiopathic adult-onset dystonia to a third body site

Ercoli, Tommaso; Erro, Roberto; Fabbrini, Giovanni; Pellicciari, Roberta; Girlanda, Paolo; Terranova, Carmen; Avanzino, Laura; Biasio, Francesca Di; Barone, Paolo; Esposito, Marcello; Joanna, Gabriella De; Eleopra, Roberto; Bono, Francesco; Manzo, Lucia; Bentivoglio, Anna Rita; Petracca, Martina; Mascia, Marcello Mario; Albanese, Alberto; Castagna, Anna; Ceravolo, Roberto; Altavista, Maria Concetta; Scaglione, Cesa; Magistrelli, Luca; Zibetti, Maurizio; Bertolasi, Laura; Moja, Mario Coletti; Cotelli, Maria Sofia; Cossu, Giovanni; Minafra, Brigida; Pisani, Antonio; Misceo, Salvatore; Modugno, Nicola; Romano, Marcello; Cassano, Daniela; Berardelli, Alfredo; Defazio, Giovanni; Cimino, Paola; Scannapieco, Sara; Ferrazzano, Gina; Brigandì, Amelia; Habetswallner, Francesco; Pascarella, Angelo; Ialongo, Tàmara; Ramella, Marina; Mazzucchi, Sonia; Moschella, Vincenzo

doi:10.1016/j.parkreldis.2021.04.022

Cited by 11 publications

(9 citation statements)

References 27 publications

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“…In December 2020, the data on 113 patients with FDYT were extracted from the IRFMD, which included data from 410 patients with functional movement disorders, 13,16 and 125 patients with IDYT selected among the 1634 patients from the IRAD 17 . The 2 groups were similar for sex, age, and educational level but differed for disease duration, dystonia distribution, and frequency of focal dystonia, which was more frequent in the patients with IDYT (Table 1).…”

Section: Resultsmentioning

confidence: 99%

Sudden Onset, Fixed Dystonia and Acute Peripheral Trauma as Diagnostic Clues for Functional Dystonia

Ercoli

Defazio

Geroin

et al. 2021

Movement Disord Clin Pract

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Background The differentiation of functional dystonia from idiopathic dystonia may be clinically challenging. Objective To identify clinical features suggestive of functional dystonia to guide physicians to distinguish functional dystonia from idiopathic dystonia. Methods Patient data were extracted from the Italian Registry of Functional Motor Disorders and the Italian Registry of Adult Dystonia. Patients with functional and idiopathic dystonia were followed up at the same clinical sites, and they were similar in age and sex. Results We identified 113 patients with functional dystonia and 125 with idiopathic dystonia. Sudden onset of dystonia, evidence of fixed dystonia, and acute peripheral trauma before dystonia onset were more frequent in the functional dystonia group. No study variable alone achieved satisfactory sensitivity and specificity, whereas a combination of variables yielded 85% sensitivity and 98% specificity. A diagnostic algorithm was developed to reduce the risk of misclassifying functional dystonia. Conclusion Our findings extend the current diagnostic approach to functional dystonia by showing that clinical information about symptom onset, fixed dystonia, and history of peripheral trauma may provide key clues in the diagnosis of functional dystonia.

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Section: Resultsmentioning

confidence: 99%

Sudden Onset, Fixed Dystonia and Acute Peripheral Trauma as Diagnostic Clues for Functional Dystonia

Ercoli

Defazio

Geroin

et al. 2021

Movement Disord Clin Pract

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“…While many studies have focused on comparing rates of risk of spread from various parts of the body, few have focused on isolating and exploring risk factors related to the larynx specifically. 3,4,6,15 Particularly, family history of LD and its possible influence over spread has not been examined. In our study, the spread of dystonia to distal body regions was found in about one-fifth of the patients who presented with adult-onset focal LD.…”

Section: Discussionmentioning

confidence: 99%

Demographics and Clinical Characteristics Associated with the Spread of New‐Onset Laryngeal Dystonia

Ghanouni,

Jona,

Jinnah

et al. 2023

The Laryngoscope

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ObjectivesAdult‐onset idiopathic laryngeal dystonia (LD) can be associated with the risk of spread to muscles in the body. Subjects with extralaryngeal onset of dystonia have exhibited spread to the larynx. Previous studies analyze the spread of other dystonias but emphasis has not been placed on LD. The objective was to identify demographic and clinical factors contributing to the spread of dystonia to and from the larynx.MethodsData were obtained from the Dystonia Coalition (DC)‐patients from 49 international clinical centers. Clinical and demographic data was taken from 143 out of 409 patients with diagnosed LD. Patient criteria included adult‐onset LD diagnosed on exam with no co‐morbid neurologic conditions and no dystonia in other locations.ResultsAmong the 143 patients, 94 (65.7%) patients were diagnosed with focal laryngeal onset, with the remainder having extralaryngeal onset. Family history and age at study were statistically significant indicators of a patient developing laryngeal versus extralaryngeal onset of dystonia. Among the laryngeal onset group, 21 cases (22.3%) had an average time of 5.81 ± 5.79 years to spread from diagnosis, most commonly to neck (61.9%). Among extralaryngeal onset patients, mean time of larynx spread was 7.92 ± 7.737 years, most commonly to neck (22.7%).ConclusionsOur data indicates approximately a quarter of patients with laryngeal‐onset dystonia will exhibit spread. There were no demographic or clinical factors that were statistically predictive of the likelihood of spread from larynx. Patients with dystonia elsewhere in the body should be counseled on the possibility of spread to larynx, and vice versa.Level of EvidenceLevel IV Laryngoscope, 2023

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Section: Discussionmentioning

confidence: 99%

“…Also, the demographic and clinical characteristics of our sample overlapped those of ICD [ 1 ]: preponderance of female gender, mean disease duration, prevalent focal expression and association with cranial dystonia in cases of 'spreading' [ 33 , 34 ]. Unlike what occurs in ICD, we observed spreading towards the upper limbs instead of the trunk [ 1 ]. To date, in tardive dystonia the cranio-cervical phenotype is the most frequent and its phenomenology does not seem to differ from that of idiopathic dystonia [ 35 , 36 ].…”

Section: Discussionmentioning

confidence: 99%

“…It is characterized by sustained, directional, involuntary movements of the head and neck that cause abnormal posturing along with head tremor and/or pain. Most cases of CD are defined as primary or idiopathic (ICD) [ 1 ]. Acquired dystonia (ACD) can be secondary to drug exposure, toxic, vascular, neoplastic, cerebral palsy (CP), brain injuries, post-infection/inflammation, functional, according to the etiological axis of the latest consensus on dystonia classification [ 2 ].…”

Section: Introductionmentioning

confidence: 99%

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Efficacy and safety of long-term botulinum toxin treatment for acquired cervical dystonia: a 25-year follow-up

Petracca

Monaco²,

Ialongo³

et al. 2022

J Neurol

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Botulinum toxin A (BoNT/A) is the first-line treatment for idiopathic cervical dystonia (ICD) and is widely used in the clinical setting. To date, scanty data are available on the effectiveness of BoNT in treating acquired cervical dystonia (ACD). Here we present a long-term follow-up of ACD patients treated with BoNT/A that focused on safety and efficacy. The study included subjects who had received at least six treatments of three commercially available BoNT/A drugs [abobotulinumtoxinA (A/Abo), incobotulinumtoxinA (A/Inco) and onabotulinumtoxinA (A/Ona)]. Safety and efficacy were assessed based on patients' self-reports regarding adverse effects (AE), duration of improvement of dystonia and/or pain relief. Global clinical improvement was measured on a six-point scale. 23 patients with ACD were administered 739 treatments (A/Abo in 235, A/Inco in 72, A/Ona in 432) with a mean number of treatments of 31 ± 20 (range 6–76) and duration of 10 ± 6 weeks (range 2–25). The mean dose was 737 ± 292 U for A/Abo, 138 ± 108 U for A/Inco and 158 ± 80 U for A/Ona. The average benefit duration was 89 ± 26 (A/Abo), 88 ± 30 days (A/Inco), and 99 ± 55 days (A/Ona) (p = 0.011); global clinical improvement for all sessions was 4 ± 1. ANOVA one-way analysis indicated that A/Ona had the best profile in terms of duration (p < 0.05), whereas A/Abo had the best pain relief effect (p = 0.002). Side effects were reported in 9% of treatments (67/739), with ten treatments (1%) complicated by two side effects. Most side effects were rated mild to moderate; severe side effects occurred following three treatments with the three different BoNT; two required medical intervention. No allergic reactions were reported. Even after 25 years of repeated treatments, all serotypes of BoNT demonstrate positive effects in treating ACD with long-lasting efficacy and safety.

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Spread of segmental/multifocal idiopathic adult-onset dystonia to a third body site

Cited by 11 publications

References 27 publications

Sudden Onset, Fixed Dystonia and Acute Peripheral Trauma as Diagnostic Clues for Functional Dystonia

Sudden Onset, Fixed Dystonia and Acute Peripheral Trauma as Diagnostic Clues for Functional Dystonia

Demographics and Clinical Characteristics Associated with the Spread of New‐Onset Laryngeal Dystonia

Efficacy and safety of long-term botulinum toxin treatment for acquired cervical dystonia: a 25-year follow-up

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