Ocular-Oral Synkinesis Without Facial Palsy After Trauma: A Case Report.

Ocular-oral synkinesis refers to the unintended contraction of the perioral muscles during voluntary eye closure. This phenomenon can either be congenital or occasionally develop during recovery from facial palsy. In this study, we report a case of ocular-oral synkinesis without facial palsy after trauma.

Synkinesis and Communicative Participation.

Background: Facial palsy (FP) impacts verbal and nonverbal communication, but the effect of synkinesis on communicative ability is unknown. Objective: Among patients with nonflaccid FP, or synkinesis, is there a correlation between disease-specific quality-of-life and communicative ability or dysfunction? Methods: Retrospective study of a series of adult patients with unilateral synkinesis. Subjects were evaluated using the Communicative Participation Item Bank (CPIB) Short Form, Facial Clinimetric Evaluation (FaCE) scale, and Synkinesis Assessment Questionnaire (SAQ). Associations between these scales were evaluated by computing Pearson correlation coefficients. Results: A total of 69 confirmed synkinesis patients were included. Synkinesis patient mean (standard deviation) CPIB score was 20.68 (±8.27; range of scale 0-30), indicative of communication restriction. A strong correlation was observed between total CPIB and FaCE scores (r = 0.66), indicating patients with synkinesis who reported better facial function also reported greater communicative ability. There was a weak correlation between CPIB and SAQ scores (r = -0.27). Conclusion: Synkinesis is associated with significant deficits in communicative ability. Communication restrictions track strongly with the FaCE scale.

Translation, cross-cultural adaptation, and validation of the Italian version of the Synkinesis Assessment Questionnaire in individuals with peripheral facial palsy.

BACKGROUND: After peripheral facial palsy, the onset of facial synkinesis results in aesthetic disfigurement and local muscle tension or pain, with possible deterioration of patient's well-being and social participation. The availability of valid instruments to evaluate patient-reported severity of facial synkinesis is important to capture the subjective perception of facial impairment. AIM: To generate and validate an Italian version of the Synkinesis Assessment Questionnaire, a patient-reported outcome measure to assess patient-perceived severity of facial synkinesis after peripheral facial palsy. DESIGN: Observational study. SETTING: Outpatient clinic of a Rehabilitation Unit. POPULATION: Seventy-five patients with peripheral facial palsy. METHODS: Through a process of translation and cross-cultural adaptation, we generated the Italian version of the questionnaire (SAQ-IT) and administered it twice to patients with peripheral facial palsy. We evaluated the clinical severity with the Sunnybrook Facial Grading System (SFGS) and the physical and social/well-being function with the two subscales of the Facial Disability Index (FDI-PHY and FDI-SWB, respectively). RESULTS: Cronbach's alpha was 0.87. Item-total correlations ranged from 0.30 to 0.70, while inter-item correlations ranged from 0.15 to 0.82, with an average value of 0.48. Test-retest reliability showed an Intraclass Correlation Coefficient of 0.946 (95% confidence interval: 0.916-0.966). The minimum detectable change (with a 95% confidence level, MDC95) was 13.14 points. The correlation between SAQ-IT and the SFGS synkinesis subscore was rho=0.74, while that with the SFGS composite score was rho=0.25, with the FDI-PHY rho=-0.11 and with the FDI-SWB rho=-0.13. CONCLUSIONS: Our study validates the SAQ-IT in Italian-speaking individuals with peripheral facial palsy, confirming its acceptable psychometric properties, and providing the MDC95. CLINICAL REHABILITATION IMPACT: The availability of a valid instrument for the evaluation of patient-perceived severity of facial synkinesis plays an important role in the definition of tailored rehabilitative interventions after peripheral facial palsy.

Synkinetic Brow Dysfunction: Diagnosis and Treatment Algorithm.

Importance: The diagnosis and management of brow dysfunction in patients with postfacial paralysis with synkinesis can be perplexing and challenging for the treating physician. Objective: To describe a novel diagnostic and treatment algorithm for brow dysfunction in patients with postfacial paralysis with synkinesis. Design: Surgical pearls-description of novel surgical technique. Setting: A private practice. Participants: Patients who underwent the operation.

Anatomic Considerations of Perinasal Musculature for Improved Dental Show During Smile in Facial Synkinesis.

Introduction: There is an anatomic explanation for upper lip and midfacial tethering resulting in lack of motion in facial synkinesis. Objective: To measure the effect of perinasal chemodenervation on dental show in the synkinetic population and clarify the anatomic relationship of perinasal musculature. Methods: Literature search was performed on anatomy of the perinasal modiolus, and anatomic evaluation was performed through human anatomic specimen dissection. Photographic outcomes were observed in synkinetic patients receiving chemodenervation to smile antagonists with and without perinasal muscle injections and assessed through naive observer survey. Retrospective outcomes for all patients receiving perinasal chemodenervation was collected utilizing Facial Clinimetric Evaluation Scale, Sunnybrook Facial Grading System (FGS), Facial Disability Index (FDI), and the Synkinesis Assessment Questionnaire. Results: Anatomic dissections demonstrated muscular confluence spanning the nasal sidewall and upper lip tethering the soft tissue to bone. Thirty-four of 53 chemodenervation patients received perinasal Botox experiencing improvement in synkinetic symptoms of the upper lip, nose, and improved dental show as noted on paired t-test for FGS (p = 0.00096), and FDI social p = 0.015) also supported by naive observer surveys (p = 0.03). Conclusions: Human anatomic specimen dissections support a perinasal confluence of musculature with bony attachments that can be successfully treated with chemodenervation in facial synkinesis patients.

Varied presentations of congenital ocular synkinesis: do they all fit congenital cranial dysinnervation disorder spectrum?

PURPOSE: Synkinesis results from nerve miswirings and causes aberrant movements of the affected muscles. We present a series of cases of rare congenital ocular synkinesis involving the extraocular muscles and the levator palpebrae superioris and speculate the possibility of classifying these entities in the spectrum of congenital cranial dysinnervation disorder. METHODS: Records of patients with the diagnosis of congenital ocular synkinesis were analyzed retrospectively. We analyzed the sex, laterality, and complete features of the ocular motility of each patient. RESULTS: Nine patients with congenital ocular synkinesis were included. A slight predominance of women was noted. In terms of laterality, four patients had only the right eye involved, four had only the left eye, and one had both eyes involved. Notably, 55.5% were orthotropic in the primary position. The third, fourth, and sixth cranial nerves were involved in the miswiring in 100%, 44.4%, and 11.1% of the cases, respectively. CONCLUSIONS: Congenital synkinesis might present in a very eclectic and uncommon fashion. The aberrant innervation in these cases classifies them into the group of congenital cranial dysinnervation disorders.

Diagnostic limitation of laryngostroboscopy in comparison to laryngeal electromyography in synkinesis in unilateral vocal fold paralysis.

PURPOSE: In clinical practice, laryngo(strobo)scopy (LS) is still mainly used for diagnostics and management of unilateral vocal fold paralysis (UFVP), although only laryngeal electromyography (LEMG) can provide information on causes of vocal fold immobility, especially on possible synkinetic reinnervation after recurrent laryngeal nerve (RLN) injury. The goal of this retrospective study was the evaluation whether signs of synkinetic reinnervation in LS can be objectified in comparison to LEMG data. METHODS: Between 1/2015 and 2/2018, 50 patients with laryngostroboscopically suspected UVFP received routine LEMG examination. The LEMG findings were retrospectively compared with LS findings. The LEMG data analysis focused on the diagnosis of synkinetic reinnervation of the TA/LCA and/or PCA. The digital LS recordings were retrospectively re-evaluated by phoniatricians considering 22 selected laryngostroboscopic parameters. RESULTS: LEMG revealed synkinesis in 23 (46%) and absence of synkinesis in 27 (54%) patients. None of the 22 parameters showed significant association between patients with synkinetic reinnervation and LS findings. The only laryngostroboscopic parameter that was significantly associated with a silent LEMG signal compared to single fiber activity in LEMG was a length difference on the side of the UVFP (p-value 0.0001; OR 14.5 (95% CI 3.047-66.81; Sensitivity 0.5; Specificity 0.9355). CONCLUSION: Our findings show that synkinesis cannot be diagnosed using only LS. This study underlines the importance of LEMG in clinical routine for detection of laryngeal synkinesis in patients with UVFP before any further therapeutic steps are initiated to avoid later therapy failure.

Role of Body Dysmorphic Disorder in Patients With Postparalytic Facial Synkinesis.

OBJECTIVES/HYPOTHESIS: To evaluate the role of body dysmorphic disorder (BDD) in patients with postparalytic facial nerve syndrome with synkinesis (PFS). STUDY DESIGN: A single-center retrospective cohort study. METHODS: A total of 221 adults (74% women; median age: 44 years; median duration since onset of facial palsy: 1.6 years) were included. To diagnose BDD, the BDD Munich Module was used. Associations with House-Brackmann grading, Stennert index grading, Facial Clinimetric Evaluation (FaCE) survey, Facial Disability Index (FDI), general quality of life (SF-36), Beck Depression Inventory (BDI), and the Liebowitz Social Anxiety Scale (LSAS) was analyzed. RESULTS: A total of 59 patients (27%) were classified as patients with BDD. Significant associations were found between the diagnosis of BDD and female gender and lower FDI, FaCE, and SF-36 scores and higher BDI and LSAS scores. Multivariate analysis revealed BDI, FaCE total score, and FaCE social function subscore as independent factors associated with BDD. CONCLUSION: BDD was a relevant diagnosis in patients with PFS. A higher BDD level was associated with general and facial-specific quality of life and more psychosocial disabilities. Optimal treatment of PFS has to include these nonmotor dysfunctions. LEVEL OF EVIDENCE: 3 Laryngoscope, 131:E2518-E2524, 2021.

Pharmacological Treatments of Bell's Palsy in Adults: A Systematic Review and Network Meta-Analysis.

OBJECTIVE: We aimed to simultaneously compare all available medical treatments for Bell's palsy using both direct and indirect data. METHODS: The literature was searched from January 1, 1990, until March 1, 2020, with no language restrictions. Randomized clinical trials comparing pharmacological interventions were included in the current network meta-analysis. We estimated summary risk ratios (RRs), 95% credible interval (CrI), and the surface under the cumulative ranking curve (SUCRA) using network meta-analyses with random effects in a Bayesian framework. The primary outcomes were complete recovery in short-term (≤3 months) and intermediate/long-term (>3 months) after randomization. The secondary outcome was synkinesis. RESULTS: In total, 21 trials comprising 2,839 participants were retrieved. In terms of good recovery, corticosteroids plus antivirals were the most effective treatment compared to placebo, with RRs ranging between 1.25 (95% CrI: 1.10, 1.43) for the short-term and 1.26 (95% CrI: 1.11, 1.45) for the intermediate/long-term recovery. For synkinesis, only corticosteroids plus antivirals (RR 0.35; 95% CrI: 0.19, 0.65) were associated with fewer synkinesis rates than placebo. The certainty of the evidence for good recovery and synkinesis was very low-low and moderate-high, respectively. CONCLUSIONS: This network meta-analysis showed that combined therapy remains the best regimen for a good recovery outcome and the only efficacious regimen for synkinesis. More research is needed to confirm these findings. Laryngoscope, 131:1615-1625, 2021.

Crumley's Classification of Laryngeal Synkinesis: A Comparison of Laryngoscopy and Electromyography.

OBJECTIVES/HYPOTHESIS: Applying the principles of misdirected nerve regeneration to the larynx, Roger Crumley in 1989 coined the term laryngeal synkinesis (LS) which he later (2000) classified into 4 types (type I - good voice, type II - involuntary twitches and poor voice, type III - adduction during inspiration, type IV - abduction during phonation). Neurophysiological data were not available for all LS patients at that time. The current study was undertaken to utilize and test the Crumley classification for a clinical interrater comparison and, secondly, compare predicted with actual laryngeal electromyography (LEMG) results. STUDY DESIGN: Descriptive study. METHODS: Laryngoscopic and LEMG data of patients with unilateral vocal fold paralysis (VFP) of 6 months duration or longer were combined for retrospective evaluation. Forty-five data sets were available for laryngoscopic classification by two local laryngologists and by Roger Crumley. Twenty-three data sets with complete thyroarytenoid (TA) and posterior cricoarytenoid (PCA) - EMG data were used to compare predicted with actual LEMG results. RESULTS: Local laryngologists were able to classify 24 of 45, Crumley 30 of 45 cases into one of the 4 synkinesis types. There was substantial agreement between examiners (Cohens Kappa 0.66 [P < .001]). Comparison of predicted and actual LEMG data showed only moderate agreement. EMG sykinesis rates were lower in TA than in PCA and highest in Crumley type I cases. CONCLUSION: The Crumley classification is helpful in describing and understanding synkinesis. It does not always correlate predictably with actual LEMG data. A complete LEMG mapping of all intrinsic muscles may improve understanding of chronic VFP. LEVEL OF EVIDENCE: 4. Laryngoscope, 131:E1605-E1610, 2021.

French Translation and Validation of the Synkinesis Assessment Questionnaire.

Synkinesis is a distressing sequela of peripheral facial palsy (PFP). This study aimed to translate and validate the Synkinesis Assessment Questionnaire (SAQ), a reliable patient-reported outcome evaluation tool for synkinesis, in French. The SAQ was translated following a standard forward-backward translation procedure. After a cognitive debriefing with 10 PFP patients, the SAQ-F was assessed amongst 50 patients for internal consistency, known-group validity, construct validity, criterion validity, and test-retest reliability. Results demonstrated that the SAQ-F was valid, reliable, and had a unidimensional structure. The SAQ-F should be accompanied by clinician-based scales, to provide valuable additional information on the severity of synkinesis.

Synkinetic Unilateral Lower Lip Palsy: Diagnosis and Technical Considerations for Facial Reanimation.

Importance: There are no universally adopted surgical techniques to treat depressor labii inferioris (DLI) dysfunction in patients with postfacial paralysis synkinesis. We describe a novel description of this disorder and technical surgical considerations for reanimation. Objective: To describe a new classification for DLI dysfunction and a surgical option to restore a natural appearing full dentition smile. Design: Surgical pearls-description of novel surgical technique. Setting: A private practice. Participants: Patients who underwent the operation.

The Auto-eFACE: Machine Learning-Enhanced Program Yields Automated Facial Palsy Assessment Tool.

BACKGROUND: Facial palsy assessment is nonstandardized. Clinician-graded scales are limited by subjectivity and observer bias. Computer-aided grading would be desirable to achieve conformity in facial palsy assessment and to compare the effectiveness of treatments. This research compares the clinician-graded eFACE scale to machine learning-derived automated assessments (auto-eFACE). METHODS: The Massachusetts Eye and Ear Infirmary Standard Facial Palsy Dataset was employed. Clinician-graded eFACE assessment was performed on 160 photographs. A Python script was used to automatically generate auto-eFACE scores on the same photographs. eFACE and auto-eFACE scores were compared for normal, flaccidly paralyzed, and synkinetic faces. RESULTS: Auto-eFACE and eFACE scores differentiated normal faces from those with facial palsy. Auto-eFACE produced significantly lower scores than eFACE for normal faces (93.83 ± 4.37 versus 100.00 ± 1.58; p = 0.01). Review of photographs revealed minor facial asymmetries in normal faces that clinicians tend to disregard. Auto-eFACE reported better facial symmetry in patients with flaccid paralysis (59.96 ± 5.80) and severe synkinesis (62.35 ± 9.35) than clinician-graded eFACE (52.20 ± 3.39 and 54.22 ± 5.35, respectively; p = 0.080 and p = 0.080, respectively); this result trended toward significance. CONCLUSIONS: Auto-eFACE scores can be obtained automatically using a freely available machine learning-based computer software. Automated scores predicted more asymmetry in normal patients, and less asymmetry in patients with flaccid palsy and synkinesis, compared to clinician grading. Auto-eFACE is a quick and easy-to-use assessment tool that holds promise for standardization of facial palsy outcome measures and may eliminate observer bias seen in clinician-graded scales. CLINICAL QUESTION/LEVEL OF EVIDENCE: Diagnostic, III.

Laryngeal Synkinesis: A Viable Condition for Laryngeal Pacing.

Laryngeal synkinesis as a form of defective healing is the rule rather than the exception in persistent vocal fold paralysis. It typically occurs 4-6 months after the onset of the recurrent laryngeal nerve paralysis. The incidence is up to 85%. Not all laryngeal muscles need to be equally affected. Reliable evidence can only be provided by a laryngeal electromyography. Physiological co-activation of the laryngeal muscles during antagonistic maneuvers must be considered. Although synkinesis undeniably worsens the prognosis for a motion recovery, it protects the muscle fibers from degeneration. A differentiation is required between favorable synkinesis (type I according to Crumley), which does not always require further therapy in the case of unilateral paralysis, and unfavorable forms of synkinesis (type II-IV) according to Crumley, which are associated with a functionally relevant malposition of the vocal fold(s) or with vocal fold jerks. Particularly when bilateral vocal fold motion does not return, type I synkinesis can be a good prerequisite for new dynamic therapy approaches, such as laryngeal pacing. The rarely occurring type II-IV synkinesis should, whenever possible, be transformed into a more favorable type I synkinesis by selective or non-selective reinnervation at an early stage of the disease. The latter applies to expected muscle atrophy with insufficient regrowth of nerve fibers.

Syngeneic Transplantation of Rat Olfactory Stem Cells in a Vein Conduit Improves Facial Movements and Reduces Synkinesis after Facial Nerve Injury.

BACKGROUND: Posttraumatic facial paralysis is a disabling condition. Current surgical management by faciofacial nerve suture provides limited recovery. To improve the outcome, the authors evaluated an add-on strategy based on a syngeneic transplantation of nasal olfactory stem cells in a rat model of facial nerve injury. The main readouts of the study were the recording of whisking function and buccal synkinesis. METHODS: Sixty rats were allocated to three groups. Animals with a 2-mm facial nerve loss were repaired with a femoral vein, filled or not with olfactory stem cells. These two groups were compared to similarly injured rats but with a faciofacial nerve suture. Olfactory stem cells were purified from rat olfactory mucosa. Three months after surgery, facial motor performance was evaluated using video-based motion analysis and electromyography. Synkinesis was assessed by electromyography, using measure of buccal involuntary movements during blink reflex, and double retrograde labeling of regenerating motoneurons. RESULTS: The authors' study reveals that olfactory stem cell transplantation induces functional recovery in comparison to nontransplanted and faciofacial nerve suture groups. They significantly increase (1) maximal amplitude of vibrissae protraction and retraction cycles and (2) angular velocity during protraction of vibrissae. They also reduce buccal synkinesis, according to the two techniques used. However, olfactory stem cell transplantation did not improve axonal regrowth of the facial nerve, 3 months after surgery. CONCLUSIONS: The authors show here that the adjuvant strategy of syngeneic transplantation of olfactory stem cells improves functional recovery. These promising results open the way for a phase I clinical trial based on the autologous engraftment of olfactory stem cells in patients with a facial nerve paralysis.