The effectiveness of physiotherapy for patients with isolated cervical dystonia: an updated systematic review and meta-analysis.

BACKGROUND: Cervical dystonia is a movement disorder typically characterized by a patterned and twisting movement of sustained or intermittent muscle contractions. Recently, new clinical trials are emerging, highlighting the potential benefit of physiotherapy (PT) on disease outcomes. Thus, the objective of this review is to update the effectiveness of PT on cervical dystonia disease outcomes and subsequently perform a meta-analysis. METHODS: Interventional studies published in English with adult patients with isolated cervical dystonia following a physiotherapy program were included. Relevant articles were searched in PubMed (MEDLINE), Web of Science, and Scopus. Cochrane and Joanna Briggs Institute risk of bias checklists were used for quality reporting. Meta-analysis was done using Review Manager 5.3 statistical software and a pooled mean difference for pain was presented. RESULTS: Fourteen articles were included in the review and two articles were included in the meta-analysis. The meta-analysis revealed that PT intervention had a significant effect on pain reduction scale (-5.00, 95% CI -6.26, -3.74) when used as an additional therapy with botulinum toxin (BoNT) injection. Additionally, findings indicate a possible positive effect of PT disease severity, disability, and quality of life. CONCLUSIONS: Physiotherapy in addition to BoNT is recommended to decrease pain. The findings suggest a reduction of disease severity, disability, and improvement in quality of life. The variety in the type and duration of PT interventions did not allow a clear recommendation of a specific type of PT.

Torticollis as a rare, late complication of VP shunt placement in the pediatric population: a case report.

The placement of a ventriculoperitoneal (VP) shunt is a common neurosurgical, pediatric procedure with various, well-documented complications occurring both in the immediate postoperative course of the procedure and at later stages. Pediatric patients need frequent revision surgeries due to body growth as well as implant failure over the course of the years. We report a rare case of a 12-year-old patient, presenting with torticollis, 11 years after the initial placement of a VP shunt. The peripheral tube had to be surgically removed due to the severe movement limitation of the cervical spine area and the tilting of the head. This is the fourth case known to be reported with this rare complication. The calcification of the tube and the formation of a rigid scar tissue along the shunt tube, combined with the body growth, are the suspected mechanisms of this mechanical malfunction of the VP-shunt. The complication could be efficiently addressed through the surgical replacement of the peripheral tube and the transection of the scar tissue in the neck area.

Disease spectrum of torticollis in children and diagnostic flowchart: A retrospective, single-centre study.

AIM: To describe the disease spectrum of torticollis in Chinese children and to improve its diagnostic flowchart. METHODS: A retrospective analysis was conducted at the Rehabilitation Department of Beijing Children's Hospital from 2017 to 2021. Patients were diagnosed and referred based on a diagnostic flowchart of torticollis. Detailed patient data were collected from the outpatient electronic medical record system. RESULTS: A total of 2047 patients met the inclusion criteria. The top five conditions were congenital muscular torticollis (CMT) (76.6%), cerebral palsy (5.1%), ocular torticollis (4.7%), brachial plexus injury (1.9%) and atlantoaxial rotary subluxation (1.3%). CMT was most common in 0-2 year olds, cerebral palsy in 3-5 year olds, and atlantoaxial rotary subluxation in 7-12 year olds. The top five referral departments were orthopaedics, ophthalmology, otolaryngology, head and neck surgery, neurology and neurosurgery. CONCLUSIONS: The disease spectrum of torticollis in children and the diagnostic flowchart provide important references for diagnosing torticollis, which necessitates multidisciplinary collaboration.

Posterior-only approach cervical hemivertebrectomy and short fusion with pedicle screws in young children with cervical scoliosis: case report and technical note.

PURPOSE: Cervical hemivertebrae (C3-6) causing significant osseous torticollis, head tilt and facial asymmetry are rare and complicated. Cervical hemivertebrectomy (CHVE) by a posterior-only approach was never reported because it is highly risky and its efficacy remains controversial. This study is to evaluate the feasibility and preliminary clinical outcomes of posterior-only approach for CHVE and torticollis correction in young children. METHODS: Four young children aged 5-9 years old with significant torticollis caused by cervical hemivertebrae underwent deformity correction consisting of cervical pedicle screw (CPS) placement with O-arm-based intraoperative navigation, CHVE using ultrasonic bone scalpel and short-segmental posterior instrumentation and fusion. Details of this novel technique were presented. The preliminary short-term clinical and radiographic outcomes were assessed. RESULTS: On average, the operative time was 312.5 ± 49.9 min, and the surgical blood loss was 375.0 ± 150.0 ml. The structural cervical scoliosis was corrected from 31.5 ± 7.3° to 11.0 ± 4.1°, and the average correction rate was 64.9%. Head tilt was favorably corrected from 11.0 ± 4.2° to 3.5 ± 2.6°. The shoulder balance improved from 6.3 ± 1.3° to 1.5 ± 1.9°. One case with C6 CHVE had convex side radiating nerve root pain but no sign of muscle power weakness. Full recovery was achieved one month after surgery. No other complication occurred. CONCLUSIONS: CHVE by a posterior-only approach was a feasible alternative option for the treatment of congenital cervical scoliosis. It could resect the CHV effectively and achieve satisfactory torticollis correction without additional anterior access surgery. Successful CPS placement in this child population was essentially important to enable reliable osteotomy closure and firm posterior instrumentation.

Retrospective analysis of atlantoaxial rotatory fixation describing age distribution and gender ratio in children and adolescents: A preliminary report.

INTRODUCTION: Atlantoaxial rotatory fixation (AARF) in children presents with an acute onset of neck pain. Almost all cases heal within a few days of onset and are treated conservatively. Because few cases of AARF have been reported, the age distribution or gender ratio of AARF in the child population have not been described enough. In Japan, the social insurance system covers all citizens. Thus, we used insurance claims data to investigate the features of AARF. The aim of this study is to examine the age distribution, compare gender ratio and determine the recurrence proportion of AARF. METHODS: We used the JMDC database to search for claims data submitted between January 2005 and June 2017 for cases of AARF in patients aged <20 years. RESULTS: We identified 1949 patients with AARF, of which 1102 (56.5%) were male. The mean age was 98.3 ± 42.2 months and 91.6 ± 38.4 months in males and females, respectively, and males with AARF were significantly older at onset than females with AARF (p < 0.001). In both sexes, the highest frequency of AARF occurred when the patient was 6 years old. There were 121 (6.2%) cases of recurrent AARF (male: 61, 5.5%; female: 60, 7.1%), but the age differences between the sexes in these cases were not statistically significant. CONCLUSIONS: This is the first report to describe the characteristics of the study population of AARF. Males were more likely to suffer from AARF than females. Furthermore, age (in months) at AARF onset was significantly higher in males than in females. Recurrence rate was not significant in both sexes.

Stigma, coping strategies, distress and wellbeing in individuals with cervical dystonia: a cross-sectional study.

Cervical dystonia (CD) is a movement disorder which causes sustained muscle contractions in the neck leading to abnormal postures and repetitive movements. As it is a highly visible condition, people with CD can experience stigma, which may lead to unhelpful coping strategies and increased psychological distress. This study investigated whether adaptive and maladaptive coping strategies mediate the relationship between stigma and psychological outcomes in people with CD. A total of 114 adults with CD completed measures of stigma, coping, health-related quality of life (HRQOL), psychological distress (depression, anxiety, stress), and psychological wellbeing at one time point. Participants' levels of distress were high, compared to the general population. Correlational analyses showed increased stigma and maladaptive coping (e.g. substance use, behavioural disengagement) were both significantly related to increased distress, lower wellbeing and lower HRQOL, whereas higher adaptive coping (e.g. acceptance, humour) was only related to higher wellbeing. In a parallel mediation model, maladaptive coping strategies mediated the relationship between stigma and distress, HRQOL and wellbeing, but adaptive coping strategies did not. These findings suggest that maladaptive coping may play an important role in explaining the relationship between stigma and some aspects of distress and wellbeing in CD. Interventions which focus on reducing different aspects of maladaptive coping may be helpful to improve wellbeing as well as reducing stigma.

Factors Influencing the Duration of Rehabilitation in Infants with Torticollis-A Pilot Study.

Background and Objectives: Torticollis is a common pediatric condition, with an incidence of 0.3-2.0%. Studies show that an adequate, tailored, and early treatment helps 90% to 95% of children recover before the first year of life and 97% of patients recover if treatment starts before the first six months. To identify the relationships between variables considered essential in the recovery process of infants with torticollis, we included factors such as the type of torticollis, age at onset of treatment, gender, birth weight, mode of delivery, fetal position in the uterus, the presence of craniofacial deformities, regions affected by postural asymmetries, and duration of the rehabilitation program. The hypothesis of the study is that early initiation of therapy can contribute to achieving favorable outcomes in the recovery process. Material and Methods: This retrospective cohort pilot study was conducted within a rehabilitation facility, spanning a duration of 1 year. The study involved a population of 41 children aged between 0 and 6 months. The rehabilitation program consisted of the application of Vojta therapy. Each session lasted 20 min, with a frequency of three times per week. Results: A total of 41% of those who started therapy in the first 3 months of life were fully recovered after 4-6 weeks of therapy. Of infants who started therapy at 5 and 6 months of age, 15% showed no improvement in measurements from 14 to 16 weeks of age, at which point the use of a cranial orthosis was recommended, and 23% experienced a plateau in measurements from 10 to 14 weeks, requiring the use of a cervical collar in conjunction with therapy. Conclusions: The findings from the study suggest that there may be a correlation between early initiation of therapy and favorable outcomes in the recovery process. The primary factors influencing the duration of recovery were identified as the presence of body asymmetries and the age at which therapy was initiated.

[Grisel's syndrome : about two cases of a atlantoaxial (C1-C2) subluxation]. / Le syndrome de Grisel : à propos de deux cas de subluxation C1-C2.

Grisel's syndrome is a non-traumatic atlantoaxial (C1-C2) subluxation and one of the causes of torticollis in children. The subluxation occurs in the context of an infection in the ENT ("Ear Nose and Throat") region or following surgery. Diagnosis is based on clinical examination and radiological assessment. Treatment is typically medical and conservative, with surgical interventions reserved for recurrences and late presentations. We discuss here two cases of C1-C2 subluxation. The first case involves a 10-year-old child with subluxation following a rhinopharyngitis. This presentation is the classical manifestation of Grisel's syndrome. Prompt management led to correction of the subluxation using medical treatment and a soft Philadelphia collar. The second case involves a 34-year-old adult who developed posterior headaches after sphenoidotomy surgery. Grisel's syndrome is less common in adults, leading to clinical challenges and delayed diagnosis (> 1 month). Reduction combined with a halo vest treatment failed, and the patient required cervical arthrodesis.

Le syndrome de Grisel est une subluxation atlanto-axoïdienne (C1-C2) non traumatique et l'une des causes de torticolis chez l'enfant. La subluxation survient dans le cadre d'une infection de la sphère ORL ou à la suite d'une chirurgie. Le diagnostic est basé sur la clinique et l'examen radiologique. Le traitement est le plus souvent médical et conservateur. Les prises en charge chirurgicales sont limitées aux récidives et aux présentations tardives.Nous discutons ici deux cas de subluxation C1-C2. La première chez un enfant de 10 ans faisait suite à une rhinopharyngite. Cette présentation correspond à la présentation typique du syndrome de Grisel. Une prise en charge rapide a permis une correction de cette subluxation à l'aide d'un traitement médical et d'une minerve souple de type Philadelphia. Le second cas concerne une adulte de 34 ans qui a présenté des céphalées postérieures à la suite d'une chirurgie de sphénoïdotomie. Le syndrome Grisel est moins fréquent chez l'adulte, ce qui a engendré une errance clinique et un diagnostic retardé (> 1 mois). La réduction associée à un traitement par haloveste a échoué et la patiente a nécessité une arthrodèse cervicale.

Spontaneous spinal epidural hematoma in a toddler presenting with torticollis: case report and literature review.

INTRODUCTION: Spontaneous spinal epidural hematoma (SSEH) is a rare entity, especially in toddlers and infants. The nonspecificity of its presenting symptoms in children may be a source of delayed diagnosis. CASE DESCRIPTION: We report the case of a 20-month-old young boy without medical history who presented with irreducible torticollis, worsened a few days later by severe tetraplegia and respiratory distress. Spinal magnetic resonance imaging (MRI) showed a posterior epidural hematoma, extending from C3 to T1 and compressing the spinal cord. An urgent decompressive surgery via an extensive laminectomy and evacuation of the clot was performed. The patient demonstrated a partial neurological recovery on follow-up. CONCLUSION: SSEH is a rare and serious condition that may compromise the functional and vital prognosis of the patient, hence the importance of prompt diagnosis and urgent treatment.

Odd presentation of shunt malfunction: a case series and review of literature.

OBJECTIVE: Children with previous ventriculoperitoneal shunt (VPS) insertion due to hydrocephalus may refer to the hospital with various clinical complaints. Shunt malfunction is frequently diagnosed in these children necessitating shunt revision. Although increased head circumference, setting sun eye in younger children, and headache, nausea/vomiting, loss of consciousness, visual disturbance, and other signs of intracranial hypertension are common clinical manifestations of shunt malfunction, some patients may present with odd or unusual symptoms. Here, we present a series of patients with shunted hydrocephalus who presented with odd and unexpected clinical manifestations of shunt malfunction. METHODS: Eight children with shunt malfunction were enrolled in this series. The age, sex, age of shunting, etiology of hydrocephalus and management, post-shunt insertion symptoms/sign, revision surgery, outcome, and follow-up were evaluated. RESULTS: Patients were aged from 1 to 13 years (mean, 6.38 years). There were 5 males and 3 females. The odd presentation associated with shunt malfunction included facial palsy in three children, ptosis in 3 children, and torticollis and dystonia each in one child. All patients underwent shunt revision except for one patient in whom a new shunt was inserted. Follow-up showed improvement of the symptoms in all patients. CONCLUSION: In this series, we reported eight patients with unusual signs and symptoms following shunt malfunction that were successfully diagnosed and managed.

How does congenital cervicothoracic scoliosis bring about early trunk tilt and coronal imbalance during curve progression: a radiographic analysis to dissect the mechanism of proximal takeoff phenomenon.

PURPOSE: To dissect the mechanism of how congenital cervicothoracic scoliosis (CTS) drive the occurrence of early trunk tilt, namely proximal takeoff phenomenon (PTO) during curve progression. METHODS: CTS patients were stratified into case and control groups according to the presence of PTO. The radiographic deformity parameters of head-neck-shoulder complex were measured and compared between the two groups. The main risk factors for PTO were identified through multiple linear regression analysis. RESULTS: 16 CTS patients with PTO were recruited, and the non-PTO group consisted of 19 CTS patients without PTO. The average Cobb angle was 64.9 ± 19.8° in PTO group and 57.7 ± 21.9° in control group (p > 0.05). Significant difference could be observed for head shift, neck tilt, trunk inclination, apex-C7 deformity angular ratio (DAR), apex translation ratio, C6 tilt, clavicle angle (CA), radiographic shoulder height (RSH), head-neck translation and coronal balance distance (CBD) (All p < 0.05) but not head tilt (p > 0.05). Multiple linear regression analysis revealed that head shift, but not neck tilt correlated significantly with the severity of trunk inclination (ß = 0.106, p = 0.003), while apex-C7 DAR and apex translation ratio were the two factors contributing significantly to the severity of head shift (ß = 0.620, p = 0.020; ß = - 0.371, p = 0.004). CONCLUSIONS: Development and progression of head shift rather than neck tilt is a significant causative factor initiating the occurrence of trunk tilt and proximal takeoff in CTS. A higher apex-C7 DAR representing a short angular upper hemi curve and a lower apex translation ratio representing poor proximal coronal compensation are key risk factors predisposing to head shift.

Experience with the management of 2599 cases of congenital muscular torticollis and a multicenter epidemiological investigation in 17 hospitals in China.

BACKGROUND: Congenital muscular torticollis (CMT) is a common musculoskeletal disease affecting infants and young children. If CMT is not treated correctly and timely, it can lead to limited head and neck movements, head and neck deviation, and abnormal posture. In order to improve patients' symptoms and alleviate the negative impact of the disease on their lives, we are committed to exploring the treatment of CMT. METHODS: The general clinical and ultrasonographic data of 2599 children with CMT who received standardized treatment at Shenzhen Children's Hospital from 2004 to 2020 were retrospectively reviewed. According to given treatment, children with CMT were divided into the physiotherapy group, physiotherapy combined with glucocorticoid treatment group, and surgical treatment group. We divided children with CMT into local mass, uniform thickening, and atrophy according to ultrasound features. General clinical information, treatment, and ultrasound examination data in each group were compared. Additionally, electronic medical records of 2344 patients admitted due to CMT in 17 tertiary children's hospitals of China's Futang Research Center of Pediatric Development (FRCPD) from 2015 to 2019 were retrospectively analyzed. Data on sex, age, year of admission and discharge, and treatment costs during hospitalization were extracted from the first medical record pages according to the ICD codes. The data were assessed for normality using the Kolmogorov-Smirnov test. Depending on the data distribution, they were analyzed using parametric tests, such as the t-test, or non-parametric tests. Qualitative data are expressed as percentages (%) and analyzed using the chi-square or Fisher's exact probability test, with α = 0.05 as the test level. P < 0.05 was considered to be indicative of a statistically significant difference. RESULTS: Three types of CMT were defined based on sternocleidomastoid muscle ultrasound examination characteristics: local mass, uniform thickening, and atrophy. Age at first diagnosis was 69.21 ± 108.41 days in local mass type group, 216.85 ± 324.09 days in uniform thickening group, and 417.88 ± 739.05 days in atrophy- type group; while age at first physiotherapy use was 94.06 ± 206.49 days, 255.00 ± 430.62 days, 540.92 ± 1059.29 respectively. The children included in local mass type group have shown a high success rate of conservative treatment, with a rate of 7.5% of children underwent surgery. Age at first diagnosis was 112.44 ± 224.12 days in the physiotherapy group, 115.87 ± 144.86 days in the physiotherapy combined with glucocorticoid treatment subgroup, whereas the age at first physiotherapy use was 137.38 ± 312.11 and 196.91 ± 344.26 days respectively. In the observation period (2015-2019) the mean age at surgery for CMT in 17 tertiary children's hospitals of the FRCPD was 50 months. Overall, 663 children with CMT were 1-2 years of age, accounting for the largest proportion (28.3%). Followed by 417 individuals (17.8%) were 7-14 years of age, indicating that there are still more children with CMT receiving surgical treatment later. CONCLUSIONS: Early diagnosis and treatment are essential to improve the conservative treatment success rate and achieve good prognosis in children with CMT. Our team's concept for treating CMT is as follows: after diagnosing the children, we will adopt the standardized protocol of treatment, with physiotherapy combined with the injection of glucocorticoid drugs and SCM release surgery, when needed. This program has a high conservative treatment success rate and may facilitate the achievement of better prognosis and reduced teratogenicity rate.

The application of electrophysiology in the correction of spastic torticollis by triple surgery-report of 96 cases.

OBJECTIVE: To investigate the effect and value of electrophysiology in the 'triple operation' (selective excision of spastic muscles in the neck, selective resection of the posterior branch of the cervical nerve and accessory neurotomy) of spastic torticollis. METHODS: Preoperative electromyography (EMG) examination was performed on 96 patients with spastic torticollis treated in our hospital from January 2015 to December 2019. The results were used to assess the responsible muscles' primary or secondary position and the function of antagonistic muscles and to formulate a personalised surgical plan. A Cascade PRO 16-channel electrophysiological diagnostic system (produced by Cadwell, USA) was used to record the evoked EMG. Target muscles were denervated under intraoperative electrophysiological monitoring and re-examined by EMG six months later to evaluate the efficacy. RESULTS: The satisfactory rate of target muscle denervation was 95%, and the overall good rate was 79.1%. CONCLUSION: Electrophysiological examination and intraoperative application may have a positive value in the selection of the operative method, improving the rate of denervation and evaluating the prognosis of the 'triple operation'.

C2 semisegmented hemivertebra - a rare anomaly presenting with torticollis.

BACKGROUND: Cervical hemivertebrae are extremely rare anomalies which usually present with restriction of neck movements and torticollis in childhood. Hemivertebrae within the craniovertebral junction have only been reported once previously in the literature. We report a case of semisegmented C2 vertebra presenting in a young adult with cervical pain and torticollis with no other associated anomalies. CASE DESCRIPTION: A 21-year-old lady presented with a history of neck pain for the past 10 days and longstanding torticollis with head tilt to the left. Computed tomographic scan of the cervical spine revealed a semisegmented hemivertebra located laterally between the C1 lateral mass and C2 on the right side which was not associated with any other bony anomalies, scoliosis or atlantoaxial dislocation. Conservative treatment with physical therapy was the chosen therapeutic strategy. CONCLUSION: Hemivertebrae within the craniovertebral junction are exceedingly rare anomalies which may present with pain and deformity in childhood and are an important cause of congenital torticollis which may be associated with other spinal, craniofacial, cardiac or renal anomalies.

Effect of physical therapy treatment in infants treated for congenital muscular torticollis - a narrative review.

Congenital muscular torticollis is a problem that arises at birth or immediately after birth in which the sternocleidomastoid muscle is shortened on the afflicted side, leading to an ipsilateral rotated of the head and a contralateral rotation of the face and jaw. To determine the effectiveness of physical therapy treatment in infants treated for congenital muscular torticollis, relevant articles published between 2011 and 2020 were located using electronic databases. A total of 9 studies out of 24 potentially relevant articles were reviewed. All studies were randomised controlled trials with 6-8 score on the Physiotherapy Evidence Database scale (Pedro scale) which showed high quality of methodology. The studies typically found significant statistical effects in the management of congenital muscular torticollis. Additionally, most of the studies reported increased adherence to exercise as another essential advantage. Conservative physical therapy management showed positive outcomes, and early physiotherapy referral showed significant reduction in treatment duration.

Pallido-putaminal connectivity predicts outcomes of deep brain stimulation for cervical dystonia.

Cervical dystonia is a non-degenerative movement disorder characterized by dysfunction of both motor and sensory cortico-basal ganglia networks. Deep brain stimulation targeted to the internal pallidum is an established treatment, but its specific mechanisms remain elusive, and response to therapy is highly variable. Modulation of key dysfunctional networks via axonal connections is likely important. Fifteen patients underwent preoperative diffusion-MRI acquisitions and then progressed to bilateral deep brain stimulation targeting the posterior internal pallidum. Severity of disease was assessed preoperatively and later at follow-up. Scans were used to generate tractography-derived connectivity estimates between the bilateral regions of stimulation and relevant structures. Connectivity to the putamen correlated with clinical improvement, and a series of cortical connectivity-based putaminal parcellations identified the primary motor putamen as the key node (r = 0.70, P = 0.004). A regression model with this connectivity and electrode coordinates explained 68% of the variance in outcomes (r = 0.83, P = 0.001), with both as significant explanatory variables. We conclude that modulation of the primary motor putamen-posterior internal pallidum limb of the cortico-basal ganglia loop is characteristic of successful deep brain stimulation treatment of cervical dystonia. Preoperative diffusion imaging contains additional information that predicts outcomes, implying utility for patient selection and/or individualized targeting.

Torticollis in childhood-a practical guide for initial assessment.

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.

Hemi-atlas resection by combined approach: case report, literature review, and technical note.

PURPOSE: Hemivertebra within the craniovertebral junction is a rare but complex spinal deformity. Torticollis caused by hemi-atlas is extremely rare. There is no consensus on the treatment of these patients. We present our experience with one case of hemi-atlas excision and torticollis correction via a combined anterior-posterior surgical approach and short-segment fixation. METHODS: An 11-year-old girl with progressive torticollis due to hemi-atlas underwent surgery consisting of combined anterior-posterior hemivertebra resection and instrumentation and had a follow up of 20 months. Pre- and postoperative radiographic features, as well as clinical outcomes, were evaluated. RESULTS: The patient had complete recovery of torticollis at a 20-month follow-up. Radiographs showed favorable deformity correction, well-balanced coronal and sagittal alignment, and solid bony fusion. CONCLUSION: For patients with congenital cervical hemivertebra within the craniovertebral junction, combined anterior-posterior hemivertebra resection with instrumentation allows for satisfactory deformity correction and good cosmetic improvement.

A rare case of atlantoaxial rotatory fixation after posterior calvarial vault expansion surgery in a Crouzon patient.

INTRODUCTION: Atlantoaxial rotatory fixation (AARF) is a rare condition that occurs most commonly in children. The torticollis caused by AARF usually presents as abnormal posturing of the head and neck, with rotation of the chin to the opposite side. AARF in children could occur due to congenital bony malformation, minor trauma, upper respiratory tract infections (Grisel's syndrome), postoperatively after head and neck (ENT) surgery, and unknown reasons. AARF in the postoperative patient is a rare and poorly understood entity. METHODS: The authors present a case report of a 2-year-old boy with Crouzon Syndrome undergoing posterior calvarial vault expansion (PVE) surgery that developed AARF as a complication. RESULTS: The authors believe that cranial vault surgery should be considered a potential risk procedure for AARF, especially if it is done in susceptible populations (syndromic craniosynostosis patients) with other underlying sequelae (tonsillar ectopia or syringomyelia). During surgery, careful attention should be paid to maintaining a neutral alignment of the patient's cervical spine as rotatory movements under anaesthesia and muscle relaxation may be contributory factors. CONCLUSION: AARF should be suspected and investigated in children with painful torticollis after craniofacial surgery.

Factors influencing and long-term effects of manual myotomy phenomenon during physiotherapy for congenital muscular torticollis.

PURPOSE: To investigate the factors influencing and long-term effects of manual myotomy (MM) occurring during physiotherapy for congenital muscular torticollis (CMT). METHODS: We retrospectively collected the clinical data of children with CMT receiving physiotherapy between 2008 and 2018. The children were divided into manual myotomy (MM) and non-manual myotomy (NMM) groups according to whether MM occurred during treatment. We assessed physiotherapy outcomes in children with CMT using craniofacial asymmetry parameters and the Cheng-Tang rating score. By measuring the ear-eye distance, ear-nose distance, eye-mouth distance, ear-mouth distance, half-head circumference, and half-head top at two sides to evaluate craniofacial asymmetry. Based on the Cheng-Tang assessment criteria, we recorded the range of rotation, range of lateral flexion, the status of the contracted muscle, the hardness of the mass, the extent of head tilting during activities and sleeping, the status of daily activities, face size, type of head shape, cranial changes, and subjective head tilting to assess the effectiveness of treatment. Clinical data and outcome indicators (craniofacial asymmetry parameters and Cheng-Tang rating score) were compared. RESULTS: The MM group had a significantly higher total Cheng-Tang rating score than the NMM group (P < 0.05). Age at initial physiotherapy session was the risk factor for MM during physiotherapy. CONCLUSION: Children with CMT developing MM during physiotherapy generally have a good outcome, although we do not recommend MM as a goal of treatment. Physiotherapists should understand this phenomenon, assess relevant factors to predict risk, and carefully observe treatment to prevent possible complications.