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Background Henry Meige (1866-1940), a French neurologist, the pupil of Charcot, is remembered for the eponym, the Meige syndrome, describing the clinical picture of craniocervical dystonia. This historical review highlights the controversies from his essay on “Le Juif Errant” (the Wandering Jew), and the evolution of his understanding of the movement disorders of the face and neck at the time of the encephalitis lethargica. Results His thesis reported 5 patients from Eastern Europe, presenting with functional neurological disorders following traumatic life experiences. He wrote with Feindel the first book on movement disorders “Les tics et leur traitement”. He pioneered the concept of focal dystonia and distinguished the facial median spasm as a dystonic movement disorder of the face. He highlighted the co-existence of psychopathology and the influence of the mental on tics and dystonia. He coined with Brissaud and Feindel the term “geste antagoniste” in cervical dystonia. He emphasized the importance of self-management and psycho-motor retraining for focal dystonia. Conclusion Meige made an invaluable contribution to our understanding of movement disorders, during his long medical career. The eponym Meige syndrome should be retained to describe an individual clinical entity.

Torticollis is encountered often in the paediatric setting and should be considered a presenting symptom, rather than a diagnosis. Aetiologies of torticollis are numerous, and the nomenclature describing underlying diagnosis can be confusing. Furthermore, children with torticollis typically present in the first instance to primary or secondary care rather than to the subspecialist. These factors can contribute to erroneous treatment of this patient-group which could be time critical in some instances. In this review, we discuss the common causes for torticollis and propose a simple clinical assessment tool and early management scheme that will assist in the differential-diagnosis and treatment pathway of this challenging condition.Conclusion: Torticollis can be the initial presentation of various conditions. The diagnosis and management tools provided in this article can aid in guiding paediatricians as to the correct initial management, imaging, and specialist referral. What is Known: • Torticollis in childhood is a very common presenting symptom with numerous aetiologies. • Management is complex, requires multiple clinical and imaging examinations, and is usually performed by non-specialized professionals. What is New: • A new, simple clinical-assessment tool under the acronym PINCH designed to aid paediatric general practitioners in diagnosing correctly the aetiology of torticollis. • A practical management scheme to aid in the treatment pathway of children with torticollis.

In the last 3 decades, the incidence of positional cranial deformations in infants, such as positional plagiocephaly and positional brachycephaly, has increased. The deviating shape often causes parental concern for the later psychosocial wellbeing of the child. Treatment options are nonoperative, varying from positional change to helmet therapy, of which the effect has often been debated. Multiple risk factors have been associated with an increased risk on the development of these deformations. The goal of this study was to assess the impact of known risk factors on the type and severity of resulting positional cranial deformation.Parents were asked to fill out a questionnaire regarding the presence of risk factors, such as gender, age, pregnancy duration, method of delivery and reasons for atypical deliveries, breech position, birth weight, developmental status, positional preference, family history, number of siblings, and torticollis presence. Treatment methods were documented and plagiocephalometry was used to measure the cranial proportions. All children were invited to participate in short-term follow-up. The significance of the risk factors and the effect of different kinds of therapy are discussed. Although no significant correlation was found between severity and risk factors, some risk factors could be correlated with the kind of positional cranial deformation. Of the different therapies, helmet therapy had a significant impact on the reduction of positional plagiocephaly.

Botulinum toxin type A (BoNTA) products are widely used for therapeutic and aesthetic indications, but there is a need for longer-lasting treatments that maintain symptom relief between injections and reduce the frequency of re-treatment. DaxibotulinumtoxinA for Injection (DAXI) is a novel BoNTA product containing highly purified 150-kDa core neurotoxin and is the first to be formulated with a proprietary stabilizing excipient peptide (RTP004) instead of human serum albumin. The positively charged RTP004 has been shown to enhance binding of the neurotoxin to neuronal surfaces, which may enhance the likelihood of neurotoxin internalization. DAXI produces robust, extended efficacy across both aesthetic and therapeutic indications. In an extensive glabellar lines clinical program, DAXI showed a high degree of efficacy, a consistent median time to loss of none or mild glabellar line severity of 24 weeks, and median time until return to baseline of up to 28 weeks. In adults with cervical dystonia, DAXI at 125 U and 250 U significantly improved Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS) total scores, with a median duration of efficacy of 24 and 20 weeks, respectively, which compares favorably with the 12–14 weeks’ duration reported for approved BoNTA products. Overall, DAXI was well tolerated, and the consistent extended duration of effect suggests that DAXI has the potential to improve the management of both aesthetic and therapeutic conditions., Plain Language Summary Botulinum toxin is used to block the nerve signals that cause muscles to contract. Products containing botulinum toxin are commonly given by injection to treat muscle spasms (such as cervical dystonia, a painful condition where the neck muscles contract involuntarily) and for cosmetic treatment of frown lines. However, the effects of the currently approved botulinum toxin products typically wear off about 3–4 months after injection and so the injections must be repeated regularly. A new product called DAXI (DaxibotulinumtoxinA for Injection) has been developed. In this product, the botulinum toxin is formulated with a unique protein (called RTP004) that has been designed to help deliver the botulinum toxin to the nerve cells. Research suggests that the RTP004 protein in DAXI adheres the botulinum toxin to the nerves close to the injection site, potentially making its effect last longer. To date, DAXI has been studied in over 3800 patients. The studies have shown that DAXI is effective for treating neck spasms (cervical dystonia) and for reducing the appearance of frown lines. Importantly, the effects of DAXI lasted up to 6 months, which is longer than seen with other botulinum toxin products. The side effects seen with DAXI are consistent in nature and frequency with those seen with other botulinum toxin products. These findings suggest that DAXI can improve both medical and cosmetic treatments due to its longer-lasting effect.

Torticollis refers to a condition in which the head is persistently tilted to one side, sometimes associated with pain. Torticollis in a child can be congenital or acquired. Torticollis as an initial manifestation of an underlying demyelinating syndrome is quite rare in children. Here, we report a 7-year-old girl who presented with persistent torticollis. Neuroimaging of the brain revealed features of a demyelinating disease. Further studies did not show any evidence of multiple sclerosis. Cerebrospinal fluid was negative for antiaquaporin-4 antibodies, antimyelin oligodendrocyte glycoprotein antibodies, and oligoclonal bands. A seronegative demyelinating disorder was considered. She was treated with pulsed methylprednisolone therapy. She responded well to steroids with no progression of illness during follow-up. Torticollis was partially improved.

To evaluate the synergistic occurrence of facial asymmetry and craniofacial deformities in cases of Congenital muscular torticollis (CMT) and to assess the outcome of surgical correction on these deformities. Twenty-three cases of CMT presenting in ENT OPD from January 2015 to December 2019, 9 cases requiring surgical intervention were included. Facial asymmetry and craniofacial deformities were evaluated and quantified by measuring gaze angle, translational deformity and orbito-alar distance. All cases underwent a bipolar tenotomy using harmonic scalpel. In all cases on comparison with preoperative status there was significant improvement (p

INTRODUCTION: In dystonia, dopaminergic alterations are considered to be responsible for the motor symptoms. Recent attention for the highly prevalent non-motor symptoms suggest also a role for serotonin in the pathophysiology. In this study we investigated the dopaminergic, serotonergic and noradrenergic metabolism in blood samples of dystonia patients and its relation with (non-)motor manifestations.METHODS: Concentrations of metabolites of dopaminergic, serotonergic and noradrenergic pathways were measured in platelet-rich plasma in 41 myoclonus-dystonia (M-D), 25 dopa-responsive dystonia (DRD), 50 cervical dystonia (CD) patients and 55 healthy individuals. (Non-)motor symptoms were assessed using validated instruments, and correlated with concentrations of metabolites.RESULTS: A significantly higher concentration of 3-methoxytyramine (0.03 vs. 0.02 nmol/L, p < 0.01), a metabolite of dopamine, and a reduced concentration of tryptophan (50 vs. 53 μmol/L, p = 0.03), the precursor of serotonin was found in dystonia patients compared to controls. The dopamine/levodopa ratio was higher in CD patients compared to other dystonia groups (p < 0.01). Surprisingly, relatively high concentrations of levodopa were found in the untreated DRD patients. Low concentrations of levodopa were associated with severity of dystonia (rs = -0.3, p < 0.01), depression (rs = -0.3, p < 0.01) and fatigue (rs = -0.2, p = 0.04).CONCLUSION: This study shows alterations in the dopaminergic and serotonergic metabolism of patients with dystonia, with dystonia subtype specific changes. Low concentrations of levodopa, but not of serotonergic metabolites, were associated with both motor and non-motor symptoms. Further insight into the dopaminergic and serotonergic systems in dystonia with a special attention to the kinetics of enzymes involved in these pathways, might lead to better treatment options.

Introduction In patients with cervical dystonia (CD), pain is a major contributor to disability and social isolation and is often the main reason patients seek treatment. Surveys evaluating patient perceptions of their CD symptoms consistently highlight pain as a troublesome and disabling feature of their condition with significant impact on daily life and work. Areas covered In this article, the authors review the epidemiology, assessment, possible mechanisms and treatment of pain in CD, including a meta-analysis of randomized controlled trial data with abobotulinumtoxinA. Expert opinion Mechanisms of pain in CD may be muscle-based and non-muscle based. Accumulating evidence suggests that non-muscle-based mechanisms (such as abnormal transmission and processing of nociceptive stimuli, dysfunction of descending pain inhibitory pathways as well as structural and network changes in the basal ganglia, cortex and other areas) may also contribute to pain in CD alongside prolonged muscle contraction. Chemodenervation with botulinum toxin is considered the first-line treatment for CD. Treatment with botulinum toxin is usually effective, but optimization of the injection parameters should include consideration of pain as a core symptom in addition to the motor problems.

Acquired cervical scoliosis previously has been reported in dogs as a clinical sign associated with Chiari‐like malformation and syringomyelia but has not been described with inflammatory central nervous system disease. A 9‐month‐old Flat‐Coated Retriever was presented with an acute onset of cervical scoliosis with no other neurological deficits. Magnetic resonance imaging identified a focal, poorly defined intramedullary lesion within the cranial cervical spinal cord. Cerebrospinal fluid (CSF) analysis indicated mononuclear pleocytosis consistent with a diagnosis of meningomyelitis of unknown etiology. A second dog, a 3‐year‐old female spayed German Shepherd, developed an acute onset of cervical scoliosis with mild generalized proprioceptive ataxia 2 months after commencing immunosuppressive corticosteroid treatment for presumed steroid‐responsive meningitis‐arteritis. Magnetic resonance imaging at the time of diagnosis disclosed a similar intramedullary lesion within the cranial cervical spinal cord, with a neutrophilic pleocytosis on CSF analysis. Both dogs were treated with immunosuppressive dosages of prednisolone, along with cytosine arabinoside in the first dog, with resolution of cervical scoliosis seen in both. To our knowledge, this is the first report of acute onset acquired, reversible cervical scoliosis in dogs with presumed immune‐mediated meningomyelitis.

Background Cervical dystonia (CD) is the most common form of focal dystonia with involuntary movements and postures of the head. The pathogenesis and neural mechanisms underlying CD have not been fully elucidated. Methods Twenty-seven newly drug-naive patients with CD and 21 healthy controls (HCs) were recruited with clinical assessment and resting-state functional magnetic resonance imaging (rs-fMRI) scanning. Severity of CD was measured by Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS) and Tsui scores. Whole-brain voxel-wise intrinsic connectivity (IC) and seed-based functional connectivity (FC) analyses were performed for detection of changes in the CD group relative to HCs, controlling for age, gender, and global time series correlation, followed by correlation analyses of IC, seed-based FC and clinically relevant features, respectively. Results In comparison with HCs, CD patients showed significantly increased IC measurement in the anterior part of the left supramarginal gyrus and extended to the inferior left postcentral gyrus (AL-SMG/IL-PCG). With this cluster as a seed, decreased FC was found in the right precentral and postcentral gyrus. Moreover, the regional IC value in the AL-SMG/IL-PCG was significantly positively correlated with TWSTRS-1 (severity) score, and significantly negatively correlated with the associated seed-based FC strength. Conclusions Our results showed signs of both hyper- and hypo-connectivity in bilateral regions of the sensorimotor network related to CD. The imbalance of functional connectivity (both hyper- and hypo-) may hint both overloading and disrupted somatosensory or sensorimotor integration dysfunction within the sensorimotor network underlying the pathophysiology of CD, thus providing a network target for future therapies.

Deep brain stimulation (DBS) of the globus pallidus internus (GPi) has become an accepted treatment for severe cervical dystonia (CD). Assessment of therapeutic efficacy of DBS mostly focused on head position at rest but hardly on limitations of head and neck mobility, which represent a functionally important impairment in CD. We aimed to determine prospectively head and neck range of motion (ROM) preoperatively and during chronic bilateral GPi DBS in a series of 11 patients with idiopathic CD or segmental dystonia with prominent CD using a computerized motion analysis. Maximum horizontal rotation of the head in the transverse plane and lateral inclination in the frontal plane were measured preoperatively and at a median of 7 months of chronic GPi DBS, using an ultrasound-based three-dimensional measuring system combined with surface electromyography of cervical muscles. Horizontal rotation of the head increased from 78.8° ± 31.5° (mean ± SD) preoperatively to 100.7° ± 24.7° with GPi DBS (p

Introduction. The relevance of the feet planovalgus deformity problem is caused by its prevalence and tendency to progression, insufficient knowledge of a number of aspects, and the complexity of treatment. Another urgent pediatric orthopedics problem is the placement torticollis in infants, and the possible relationship of this problem with feet planovalgus deformity. Among the least studied aspects of these diseases categories it is necessary to highlight the problem of the peculiarities of the osteopathic status in children with feet planovalgus deformity with a history of placement torticollis, and the dynamics of clinical manifestations during their osteopathic correction.The aim of the research was to study the features of the osteopathic status in patients with planovalgus deformity of the feet in combination with a history of placement torticollis and without such a combination, and to evaluate the clinical efficacy of complex therapy, including osteopathic correction.Materials and methods. The study involved 60 patients aged 7–14 years with planovalgus deformity of the feet. The study participants were divided into two groups. The first group included 30 patients with planovalgus deformity of the feet and a history of placement torticollis. The second group included 30 patients with planovalgus deformity of the feet, who did not have a history of torticollis. All study participants received complex therapy (orthopedic treatment and osteopathic correction). At the beginning and at the end of the course of osteopathic correction, the condition of the arch of the foot (the severity of fl at feet) and osteopathic status were assessed in all study participants.Results. In the first group there were prevailed somatic dysfunctions (SD) of the head region, the detection rate was 86%, the pelvic region (somatic component — C), 76 %, and the neck region (C), 13 %. Among the second group participants there were prevailed SD of the pelvic region (C) — the detection rate was 100 %, the lower extremities, 43 %, and the lumbar region (C), 13 %. The SD in the pelvic region was the most typical; however, more often (ppppppConclusion. The revealed differences between the participants of the groups in the prevalence and number of regional dysfunctions and the prevalence of local SD could, presumably, reflect the relationship between the placement torticollis and planovalgus deformity of the feet. Based on the absence of differences in the degree of severity of flat feet between the participants of the groups, it can be assumed that the history of the placement torticollis is not associated by itself with the aggravation of the severity of feet planovalgus deformity.

Introduction Isolated head tremor, a pathological condition characterized by head tremor without dystonic postures or tremor in other body parts, has recently been suggested to be a form of dystonia. It is however still unclear whether isolated head tremor precedes dystonia or remains unmodified overtime. Methods We enrolled 20 patients with isolated head tremor. For each patient, we assessed videos recorded at enrollment and after 5 years. The videotapes were reviewed by two independent experienced movement disorder specialists who evaluated and scored tremor and CD severity using the Fahn-Tolosa-Marin Clinical Rating Scale for Tremor and the revised Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS), respectively. Results Upon enrollment, all 20 patients showed isolated head tremor. Mean tremor severity was 2.7 ± 0.9 as measured using the Fahn-Tolosa-Marin Clinical Rating Scale for Tremor total score. At the 5-year follow-up examination, 15 (75%) of the 20 patients with isolated head tremor showed dystonic postures in the neck, while the remaining 5 patients (25%) had only isolated head tremor. Mean severity of dystonia as measured using the TWSTRS-2 total score was 11.8 ± 3.6. Head tremor severity was unchanged between baseline and the 5-year follow-up examination (p > 0.05). At the follow-up examination, no patients had tremor or dystonia in a body part other than the neck, nor did they develop bradykinesia or other parkinsonian signs. Conclusions Our longitudinal study demonstrated that patients with isolated head tremor may develop cervical dystonia over time.

Case report. Juvenile rheumatoid arthritis (JRA) typically presents with fever, rash, anterior uveitis, and/or joint pain. We present three cases with initial torticollis due to rotatory subluxation of C1–C2 as an initial sign of JRA. Three girls, ages 5–9, presented with C1-2 rotatory subluxation. Traction was able to reduce the atlanto-axial joint in all cases. Based on imaging, history, exam, and laboratory results, they were diagnosed with JRA. After reduction of the atlantoaxial joint, they were transitioned to a halo vest and disease-modifying antirheumatic drugs (DMARDs). The older 2 children underwent C1-2 fusion. The younger child has minimal symptoms and has not undergone surgical intervention 4 years from initial presentation. Rotatory subluxation can be the first presenting sign of JRA. Younger children may be able to be treated conservatively with traction and medication, while older children may require occiput to C2 fusion due to bony destruction and basilar invagination. IV.

Background The dentatorubrothalamic tract (DRTT) remains understudied in idiopathic cervical dystonia (CD), despite evidence that the pathway is relevant in the pathophysiology of the disorder. Objective The aim of this study was to examine the DRTT in patients with CD using diffusion tensor imaging (DTI)-based tractography. Methods Magnetic resonance imaging scans from 67 participants were collected to calculate diffusion tractography metrics using a binary tractography-based DRTT template. Fractional anisotropy and diffusivity measures of left and right DRTT were computed and compared between 32 subjects with CD and 35 age-matched healthy volunteers. Results Fractional anisotropy of right DRTT and mean and axial diffusivity of left DRTT were significantly reduced in patients with CD. Similar abnormalities were observed in patients with focal CD and patients with CD without tremor. DTI metrics did not correlate with disease duration or severity. Conclusions Significant reductions in DTI measures suggest microstructural abnormalities within the DRTT in CD, characterized by a tractography pattern consistent with decreased axonal integrity. © 2021 International Parkinson and Movement Disorder Society.

Cervical Dystonia (CD) and Parkinson's disease, particularly tremor-dominant motor phenotype (TD-PD), showed a selective deficit of time-based prospective memory (TBPM). The two movement disorders are mainly characterized by dysfunctions of basal-ganglia and prefrontal cortex but it is reported that cerebellum also plays a key role in their pathogenesis. These cerebral structures are specifically involved in TBPM rather than in event-based PM (EBPM), but until now no study directly compared these two components of PM between CD and TD-PD patients. Therefore, the present study aimed at investigating if differences in PM functioning between CD and TD-PD patients might exist and if the type of movement disorder moderated the relationship between deficit of PM and deficit of executive functions and retrospective memory. Thirty TD-PD, 27CD patients and 29 healthy subjects (HCs), matched for demographic features, underwent neuropsychological tests for PM, executive functions, retrospective memory and self-rated questionnaires. The three groups did not differ on neuropsychological variables except for TBPM where TD-PD and CD patients performed worse than HCs; moreover, TD-PD performed worse than CD patients. Moderation analysis indicated that the type of movement disorder moderated the relationship between executive dysfunction and TBPM, but not EBPM. In conclusion, selective deficit of TBPM characterizes both CD and TD-PD but it is associated with executive dysfunction only in TD-PD. It might be possible to speculate that the involvement of the cerebellum, responsible for internal timing processes, could explain the impairment of TBPM in both movement disorders. This issue deserves to be explored in future neuroimaging studies.

Grisel’s syndrome (GS) is a rare syndrome which refers only to non-traumatic atlantoaxial subluxation. This syndrome predominantly occurs in young children following an upper respiratory infection or otolaryngologic procedures. An eight-year-old girl with a delayed diagnosis of GS was admitted to our outpatient clinic with complaints of painful torticollis and neck stiffness. Three-dimensional computed tomography revealed rotatory atlantoaxial subluxation. After consulting with the neurosurgery department, the patient underwent surgery. The significance of this patient was that she was unable to be diagnosed early and atlantoaxial subluxation remained hidden for five years without any complications. In conclusion, this rare case highlights the importance of delayed diagnosis of GS and clinicians should be aware of this syndrome.

Objective: The treatment of C1-C2 rotatory dislocation remains controversial and surgery is rare. Surgical treatment is indicated when the injury satisfies the instability criteria or when it cannot be reduced. The objective of this study is to analyze the principles and the adaptations necessary for treating these injuries in the pediatric population. Methods: A retrospective case series study. Three cases of patients diagnosed with traumatic C1-C2 rotatory dislocation and treated surgically in our hospital were studied. Through critical analysis of the available literature, a practical guide was proposed to establish the principles and competencies for the treatment of these injuries. Results: The operated cases were female patients between 8 and 16 years of age, with a diagnosis of traumatic atlantoaxial dislocation. Two patients required preoperative skeletal traction with halo. All patients underwent posterior instrumented arthrodesis, two with a transarticular screw technique and one with mass and C2 isthmic (Göel-Harms) screws. Conclusion:. It is essential to determine if the injury is stable and reducible. We recommend treating this type of injury keeping the criteria and competencies related to the stability, alignment, biology and function of the spine in mind. Level of evidence IV; Case series.

Dystonia is conceptualized as a network disorder involving basal ganglia, thalamus, sensorimotor cortex and the cerebellum. The cerebellum has been implicated in dystonia pathophysiology, but studies testing cerebellar function in dystonia patients have provided equivocal results. This study aimed to further elucidate motor network deficits in cervical dystonia with special interest in the role of the cerebellum. To this end we investigated motor learning tasks, that differ in their dependence on cerebellar and basal ganglia functioning. In 18 cervical dystonia patients and 18 age matched healthy controls we measured implicit motor sequence learning using a 12-item serial reaction time task mostly targeting basal ganglia circuitry and motor adaptation and eyeblink conditioning as markers of cerebellar functioning. ANOVA showed that motor sequence learning was overall impaired in cervical dystonia (p = 0.01). Moreover, unlike healthy controls, patients did not show a learning effect in the first part of the experiment. Visuomotor adaptation and eyeblink conditioning were normal. In conclusion, these data lend support to the notion that motor learning deficits in cervical dystonia relate to basal ganglia-thalamo-cortical loops rather than being a result of defective cerebellar circuitry.

In cervical dystonia, functional MRI (fMRI) evidence indicates changes in several resting state networks, which revert in part following the botulinum neurotoxin A (BoNT) therapy. Recently, the involvement of the cerebellum in dystonia has gained attention. The aim of our study was to compare connectivity between cerebellar subdivisions and the rest of the brain before and after BoNT treatment. Seventeen patients with cervical dystonia indicated for treatment with BoNT were enrolled (14 female, aged 50.2 ± 8.5 years, range 38–63 years). Clinical and fMRI examinations were carried out before and 4 weeks after BoNT injection. Clinical severity was evaluated using TWSTRS. Functional MRI data were acquired on a 1.5 T scanner during 8 min rest. Seed-based functional connectivity analysis was performed using data extracted from atlas-defined cerebellar areas in both datasets. Clinical scores demonstrated satisfactory BoNT effect. After treatment, connectivity decreased between the vermis lobule VIIIa and the left dorsal mesial frontal cortex. Positive correlations between the connectivity differences and the clinical improvement were detected for the right lobule VI, right crus II, vermis VIIIb and the right lobule IX. Our data provide evidence for modulation of cerebello-cortical connectivity resulting from successful treatment by botulinum neurotoxin.

Torticollis is a clinical diagnosis with heterogeneous causes. We present an unusual case of acquired torticollis in an 8-month-old female infant with a large cerebellopontine angle arachnoid cyst. Symptoms resolved after surgical fenestration. Non-traumatic acquired or new-onset torticollis requires brain imaging, and posterior fossa lesions are an important entity in the differential for pediatric clinicians.

Acquired torticollis is not a separate disease rather a warning sign of an underlying disorder. The etiology of torticollis in children is quite wide. Hematogenous osteomyelitis of the first cervical vertebra (the atlas) is an extremely rare and potentially dangerous pathology. The aim is to acquaint physicians of different specialties with an extremely rare condition – acute hematogenous osteomyelitis of the atlas. Case Report. A case of osteomyelitis of the first cervical vertebra in a 7-year-old boy is presented. An important sign of the disease is torticollis. Conclusions. Hematogenous osteomyelitis of the atlas is an extremely rare and potentially dangerous pathology. Regardless of its incidence, it must be considered when examining a child with acquired wryneck. All the ENT specialists must possess the knowledge of this disease, as they are most likely the first doctors to see such a child. The research was carried out in accordance with the principles of the Helsinki Declaration. The informed consent of the patient was obtained for conducting the studies. No conflict of interest was declared by the authors. Key words: torticollis, osteomyelitis of the atlas, C1 osteomyelitis, vertebral osteomyelitis, children.

OBJECTIVEThe primary treatment for atlantoaxial rotatory fixation (AARF) remains controversial. The aim of this study was to investigate the primary treatment for AARF and create an algorithm for primary treatment.METHODSThe authors analyzed the data of 125 pediatric patients at four medical institutions from April 1989 to December 2018. The patients were reported to have neck pain, torticollis, and restricted neck range of motion and were diagnosed according to the Fielding classification as type I or II. As a primary treatment, 88 patients received neck collar fixation, and 28 of these patients did not show symptom relief and required Glisson traction. Thirty-seven patients were primarily treated with Glisson traction. In total, 65 patients, including neck collar treatment failure patients, underwent Glisson traction in hospitals.RESULTSThe success rate of treatment was significantly higher in the Glisson traction group (97.3%) than in the neck collar fixation group (68.2%) (p = 0.0001, Wilcoxon test). In the neck collar effective group, Fielding type I was more predominant (p = 0.0002, Wilcoxon test) and the duration from onset to the first visit was shorter (p = 0.02, Wilcoxon test) than that in the neck collar ineffective group. Using multivariate logistic regression analysis with the above items, the authors generalized from the estimated formula: logit [p(success group by neck collar fixation group)|duration from onset to the first visit (x1), Fielding type (x2)] = 0.4(intercept) − 0.15x1 + 1.06x2, where x1 is the number of days and x2 = 1 (for Fielding type I) or −1 (for Fielding type II). In cases for which the score is a positive value, the neck collar should be chosen. Conversely, in cases for which the score is a negative value, Glisson traction should be the first choice.CONCLUSIONSAccording to this formula, in patients with Fielding type I AARF, neck collar fixation should be allowed only if the duration from onset is ≤ 10 days. In patients with Fielding type II, because the score would be a negative value, Glisson traction should be performed as the primary treatment.

The article presents the ranked risk factors for the development of torticollis in children. We conducted a study of 72 children with a diagnosis of torticollis at the age of 3 months to 17 years of age, who were treated at the National Center for Maternal and Child Health (Kyrgyz Republic). Significant risk factors for the birth of children with torticollis were toxicosis and transverse presentation of the fetus, followed by the threat of miscarriage and childbirth with stimulation.

Background Although several brain networks play important roles in cervical dystonia (CD) patients, regional homogeneity (ReHo) changes in CD patients have not been clarified. We investigated to explore ReHo in CD patients at rest and analyzed its correlations with symptom severity as measured by Tsui scale. Methods A total of 19 CD patients and 21 gender-, age-, and education-matched healthy controls underwent fMRI scans at rest state. Data were analyzed by ReHo method. Results Patients showed increased ReHo in the right cerebellum crus I and decreased ReHo in the right superior medial prefrontal cortex (MPFC). Moreover, the right precentral gyrus, right insula, and bilateral middle cingulate gyrus also showed increased ReHo values. A significantly positive correlation was observed between ReHo value in the right cerebellum crus I and symptom severity (p Conclusions Our investigation suggested abnormal ReHo existed in brain regions of the “pain matrix” and salience network (the right insula and bilateral middle cingulate gyrus), the motor network (the right precentral gyrus), the cerebellum and MPFC and further highlighted the significance of these networks in the pathology of CD.

Sternocleidomastoid (SCM) pseudotumors, also known as fibromatosis colli or congenital torticollis, are painless benign neck lumps found in newborns. Whilst unilateral cases are relatively common, bilateral SCM pseudotumors are a rare phenomenon with only a handful of cases reported internationally. We present the case of a 5-week-old infant who was brought to the emergency department with painless, bilateral, palpable anterior neck masses following a slightly traumatic but otherwise uncomplicated spontaneous delivery. An ultrasound scan of his neck revealed well-defined soft tissue lesions within both of the SCM muscles. He was subsequently diagnosed with bilateral SCM pseudotumors. This case emphasizes the importance of considering this entity as a differential diagnosis in infants presenting with bilateral palpable neck masses.

Background To observe the characteristics of binocular integration and stereopsis in children with television torticollis. Methods A retrospective study was carried out, where data were collected from 25 children with television torticollis as the disease group after refractive error correction and 25 normal children as the control group. A virtual reality system was used to assess and analyze the characteristics of binocular integration by a contrast balance test and binocular stereopsis. Results The 25 children in the disease group included 17 males and 8 females with an average age of 7.5 ± 1.9 years old and an average binocular spherical equivalent of − 0.35 ± 1.46D. The 25 children in the control group were also 17 males and 8 females with an average age of 7.3 ± 2.2 years old and the average binocular spherical equivalent of − 0.48 ± 0.93D. No significant differences were found in the horizontal bar contrast balance test between the 2 groups at near and far distances. Near-distance vertical bar contrast balance test was normal in 23 subjects and suppressed in 2 subjects in the control group, while it was normal in 13 subjects and suppressed in 12 subjects in the disease group, which showed a statistically significant difference (P = 0.002). Far distance vertical bar contrast balance test was normal in 24 subjects and suppressed in 1 subject in the control group, normal in 7 subjects and suppressed in 18 subjects in the disease group, showing a statistically significant difference (P = 0.000). All subjects in the 2 groups showed 100〞 as near distance stereoacuity. At far distance, the mean stereoacuity was 176.00〞 ± 92.56〞 in the control group, and 352.00〞 ± 270.99〞 in the disease group, with a statistically significant difference (P = 0.011). Conclusion By using virtual reality technology, defects in binocular visual function were found in children whose television torticollis persisted after regular refractive error correction. Television torticollis may be associated with the deficit of binocular integration for vertical bars and far distance stereopsis.

In a recent workshop on "Defining research priorities in dystonia,", there was absolutely no reference to sex as a factor in disease pathogenesis. In this viewpoint paper, we argue that the most distinctive aspects of adult onset isolated focal dystonia are the marked sex-related differences demonstrated by epidemiological, clinical, and laboratory studies in patients with adult onset dystonia, particularly in cervical dystonia, the most common presentation. We propose that the future focus of research should be on neurobiological mechanisms underlying the profound sexual dimorphism in this disorder. Targeting research into gamma aminobutyric acid (GABA)ergic function, which also shows similar sexual dimorphism, would be most productive in elucidating the pathogenesis of adult onset dystonia. © 2021 International Parkinson and Movement Disorder Society.