While congenital muscular torticollis (CMT) can occur along with other conditions, such as clavicle fracture or brachial plexus injury, these conditions exist outside the sternocleidomastoid muscle (SCM). We present a rare case with concurrence of CMT and a malignant tumor inside the same SCM, along with serial clinical and radiological findings of the atypical features of CMT. The malignant tumor was in fact a low-grade fibromyxoid sarcoma. To the best of our knowledge, the current case is the first of a concurrent condition of CMT inside the SCM. This case suggests that concurrent conditions could exist either inside or outside the SCM with CMT. Therefore, a thorough evaluation of SCM is required when subjects with CMT display atypical features, such as the increase of mass or poor response to conservative therapy. In that case, appropriate imaging modalities, such as ultrasonogram or magnetic resonance imaging, are useful for differential diagnosis.
Citations
Grisel syndrome is a rare, non-traumatic atlanto-axial subluxation associated with an inflammatory or infectious process in the upper neck. According to the two-hit hypothesis, which is widely accepted for the pathogenesis of Grisel syndrome, preexisting ligamentous laxity of the atlanto-axial joint is regarded as the first hit. An inflammatory or infectious process of the atlanto-axial joint acts as the second hit, resulting in non-traumatic atlanto-axial subluxation. We report on a 6-year-old girl with atlanto-axial subluxation following retropharyngeal and cervical lymphadenitis. She was diagnosed with Grisel syndrome, for which an initial computed tomography did not show any preexisting ligamentous laxity of the atlanto-axial joint. A literature review found only 4 case reports on Grisel syndrome with an initially normal atlanto-axial joint. The present case offers some evidence that a single hit, such as inflammatory changes in the atlanto-axial joint, might cause Grisel syndrome, even without underlying ligamentous laxity.
Citations
To identify the correlation between change in spinal deformities after surgical release and age at the time of surgery, and the effectiveness of surgical release in patients with neglected congenital muscular torticollis (CMT).
This was a retrospective study of 46 subjects with neglected CMT who had undergone surgical release at age ≥5 years at a tertiary medical center between January 2009 and January 2014. Spinal deformities were measured on anteroposterior plain radiographs of the cervical and whole spine, both preoperatively and postoperatively, to assess 3 parameters: cervicomandibular angle (CMA), lateral shift (LS), and Cobb angle (CA). We analyzed the change in spinal deformities after surgical release in consideration of age at the time of surgery.
The median age at the time of surgery was 12.87 years. All 3 parameters showed significant improvement after surgical release (median values, pre- to post-surgery: CMA, 12.13° to 4.02°; LS, 18.13 mm to 13.55 mm; CA, 6.10° to 4.80°; all p<0.05). There was no significant correlation between age at the time of surgery and change in CMA (R=0.145, p=0.341) and LS (R=0.103, p=0.608). However, CA showed significant improvement with increasing age (R=0.150, p=0.046).
We assessed the correlation between change in spinal deformities after surgical release and age at the time of surgery. We found that that surgical release is effective for spinal deformities, even in older patients. These findings enhance our understanding of the effectiveness and timing of surgical release in patients with neglected CMT.
Citations
To evaluate the clinical usefulness of sonoelastography in infants with congenital muscular torticollis (CMT).
The medical records of 215 infants clinically diagnosed with CMT were retrospectively reviewed. Fifty-three infants met the inclusion criteria as follows: 1) infants diagnosed as CMT with a palpable neck mass before 3 months of age, 2) infants who were evaluated initially by both B-mode ultrasonography and sonoelastography, and 3) infants who had received physical therapy after being diagnosed with CMT. We checked the thickness of the sternocleidomastoid (SCM) muscles in B-mode ultrasonography, strain ratio of the SCM muscles in sonoelastography, and treatment duration. We evaluated the correlation between the treatment duration and the following factors: SCM muscle thickness, ratio of SCM muscle thickness on the affected to unaffected side (A/U ratio), and strain ratio.
Both the thickness of the affected SCM muscle and the A/U ratio did not show significant correlation with the treatment duration (p=0.66, p=0.90). The strain ratio of the affected SCM muscle was significantly greater than that of the unaffected SCM muscle (p<0.001), and the strain ratio showed significant correlation with the treatment duration (p=0.001).
Sonoelastography may be a useful adjunctive tool to B-mode ultrasonography for evaluating infants with CMT, especially when predicting their rehabilitation outcomes.
Citations
To evaluate the craniofacial asymmetry in adults with neglected congenital muscular torticollis (CMT) by quantitative assessment based on craniofacial three-dimensional computed tomography (3D-CT).
Preoperative craniofacial asymmetry was measured by 3D-CT for 31 CMT subjects ≥18 years of age who visited a tertiary medical center and underwent 3D-CT between January 2009 and December 2013. The relationship between the age and the severity of craniofacial asymmetry was analyzed in reference to anteroposterior length asymmetry of the frontal bone and zygomatic arch, vertical and lateral displacements of the facial landmarks, and mandibular axis rotation.
The age at CT was 27.71±7.02 years (range, 18-44 years). All intra-class correlation coefficients were higher than 0.7, suggesting good inter-rater reliability (p<0.05) of all the measurements. The frontal and the zygomatic length ratio (i.e., the anteroposterior length asymmetry on the axial plane) was 1.06±0.03 and 1.07±0.03, respectively, which was increased significantly with age in the linear regression analysis (r2=0.176, p=0.019 and r2=0.188, p=0.015, respectively). The vertical or lateral displacement of the facial landmarks and rotation of the mandibular axis did not significantly correlate with age (p>0.05).
Craniofacial asymmetry of neglected CMT became more severe with age in terms of anteroposterior length asymmetry of the ipsilateral frontal bone and zygomatic arch on the axial plane even after growth cessation. This finding may enhance the understanding of therapeutic strategies for craniofacial asymmetry in adults with neglected CMT.
Citations
To quantify magnetic resonance imaging (MRI) findings of congenital muscular torticollis (CMT) and to demonstrate the usefulness of quantitative MRI findings in the management of CMT.
This was a retrospective study of 160 subjects with CMT who had undergone neck MRI at the age of 48 months or younger at a tertiary medical center. Among the 160 subjects, 54 had undergone surgical release of CMT and 106 subjects had not undergone surgery. For the quantitative analysis, the ratios of area and intensity of the MRI findings were measured and compared between the two groups (ratio of area = the largest cross-sectional area of the SCM with CMT - the cross-sectional area of the contralateral SCM without CMT / the cross-sectional area of the contralateral SCM without CMT; ratio of intensity = the mean gray color intensity of the contralateral SCM without CMT - the lowest mean gray color intensity of the SCM with CMT / the mean gray color intensity of the contralateral SCM without CMT). Receiver operating characteristic (ROC) curve analysis was conducted for the ratios of area and intensity in order to find the optimal cutoff value for determining the need for surgery in CMT cases.
The ratios of area and intensity were significantly higher in the surgical group than in the non-surgical group (p≤0.001), suggesting that the sternocleidomastoid muscle (SCM) was thicker and darker in the surgical group than in the non-surgical group. The optimal cutoff value for the ratio of area was 0.17 and that for the ratio of intensity was 0.05. All subjects with a ratio of intensity less than 0.03 belonged to the non-surgical group, and all subjects with a ratio of intensity greater than 0.16 were categorized in the surgical group.
The quantitative MRI findings, i.e., ratios of area and intensity, may provide a guideline for deciding the need for surgical intervention in CMT patients. Further prospective studies are required to verify these findings.
Citations
To determine which factors affect the rehabilitation duration in patients with congenital muscular torticollis (CMT) and to predict the duration of rehabilitation and prognosis.
One hundred and eighteen patients (79 males and 39 females) who were diagnosed with CMT and received physical therapy were enrolled in this study. We retrospectively reviewed the information in terms of sex, gestational age, birth weight, methods of delivery, fetal presentation, age at diagnosis, the affected sternocleidomastoid (SCM) muscle site, SCM muscle thickness, ratio of muscle thickness on the affected side to that on the unaffected side (called the 'abnormal/normal [A/N] ratio'), and range of motion for cervical rotation and side bending.
The SCM muscle thickness and A/N ratio had a positive linear relationship with the rehabilitation duration. Patients who were in the breech position needed longer rehabilitation. The birth weight and age at diagnosis were negatively correlated with the rehabilitation duration. However, the cervical range of motion, mass site, sex, gestational age, and methods of delivery were not correlated with the rehabilitation duration.
Patients with a thicker SCM, lower birth weight, and history of breech delivery had a longer rehabilitation duration.
Citations
Congenital muscular torticollis (CMT) and craniosynostosis are diseases that cause plagiocephaly and craniofacial asymmetry in children. In our literature review, we did not find any report of concurrent manifestation of CMT and craniosynostosis. A 41-month-old boy visited our hospital with left torticollis, right laterocollis, and craniofacial asymmetry as the main findings. During clinical examination, prominent right sternocleidomastoid muscle and limited range of motion of the neck were noted, and right CMT was confirmed by magnetic resonance imaging of the neck. Three-dimensional computed tomography of the skull, which was conducted due to the unusual appearance of the skull with a large head circumference, mild brachycephaly, as well as left plagiocephaly, revealed premature closure of the sagittal suture. Thus, we report the first case that showed concurrence of CMT and sagittal synostosis. We recommend that concurrently manifested craniosynostosis needs to be examined if the subject with CMT displays unusual craniofacial asymmetry to a greater extent than deformational plagiocephaly.
Citations
Grisel syndrome is a condition of uncertain etiology characterized by a non-traumatic atlantoaxial subluxation following an infection in the head and neck region. Although first described in 1830, the exact pathophysiology of Grisel syndrome remains unclear. We present a case of atlantoaxial subluxation after acute lymphadenitis diagnosed with a dynamic computed tomography (CT) and magnetic resonance imaging (MRI). A previously healthy 9-year-old male patient presented with torticollis of sudden onset. Dynamic CT and MR imaging showed rotary atlantoaxial subluxation and inflammation surrounding the cervical spinal ligaments. A follow-up MRI of the cervical spine, taken 3 weeks after the onset of symptoms, showed a complete resolution of subluxation and inflammation surrounding the cervical spinal ligaments. In this case report, we support the hypothesis that an inflammation-induced laxity of the cervical ligaments is the pathologic key to Grisel syndrome using radiologic findings.
Citations
To investigate the factors affecting rehabilitation outcomes in children with congenital muscular torticollis (CMT).
We retrospectively reviewed the medical records of 347 patients who were clinically suspected as having CMT and performed neck ultrasonography to measure sternocleidomastoid (SCM) muscle thickness. Fifty-four patients met the inclusion criteria. Included were demographic characteristics as well as measurements of cervical range of motion (ROM), SCM muscle thickness, and the abnormal/normal (A/N) ratio, defined as the ratio of SCM muscle thickness on the affected to the unaffected side.
Subjects were divided into three groups depending on degree of cervical ROM (group 1A: ROM>60, n=12; group 1B: 60≥ROM>30, n=31; group 1C: ROM≤30, n=11), the SCM muscle thickness (Th) (group 2A: Th<1.2 cm, n=23; group 2B: 1.2≤Th<1.4 cm, n=18; group 2C: Th≥1.4 cm, n=13), and the A/N ratio (R) (group 3A: R<2.2, n=19; group 3B: 2.2≤R<2.8, n=20; group 3C: R≥2.8, n=15). We found that more limited cervical ROM corresponded to longer treatment duration. The average treatment duration was 4.55 months in group 1A, 5.87 months in group 1B, and 6.50 months in group 1C. SCM muscle thickness and the A/N ratio were not correlated with treatment duration.
Infants with CMT who were diagnosed earlier and had an earlier intervention had a shorter duration of rehabilitation. Initial cervical ROM is an important prognostic factor for predicting the rehabilitation outcome of patients with CMT.
Citations
To determine whether a routine ultrasonography (US) is necessary for diagnosis of developmental dysplasia of hip (DDH), presenting with congenital muscular torticollis (CMT).
Cases of 133 patients (81 males, 52 females) diagnosed as CMT were reviewed, retrospectively. We reviewed the medical charts and diagnostic examination. We also assessed the coincidence of CMT and DDH, and investigated the clinical features of CMT related to DDH.
Twenty (15.0%) patients out of 133 CMT patients were diagnosed as having DDH by US. Of whom, 8 patients were radiographically positive and 4 patients were both clinically and radiographically positive. Nine patients were treated with a harness and 1 of them needed closed reduction and casting. Out of 9 patients treated with a harness, only 4 were clinically positive. The difference and ratio of the sternocleidomastoid (SCM) muscle thickness between the normal and abnormal side was significantly greater in DDH patients (p=0.014). Further, receiver operating characteristic analysis showed when the SCM ratio is greater than 2.08 and the SCM difference is greater than 6.1 mm, the efficiency of US for the diagnosis of the DDH was found to be the best (p<0.05).
To evaluate DDH, physical examination showed low sensitivity and radiologic study has limitation for the child before 4 to 6 months of age. Therefore, we recommend that hip is screened by US for the diagnosis of DDH associated with CMT when physical examination is positive or CMT patients with large SCM difference and high SCM ratio.
Citations
(1) To present the magnetic resonance imaging (MRI) findings of congenital muscular torticollis (CMT) of subjects who underwent surgical release and subjects who showed a good prognosis with stretching exercises and (2) to correlate the MRI findings with the histopathologic findings of CMT for subjects who underwent surgical release in order to examine the hypothesis that the MRI findings of CMT can be used as a determinant to perform surgical release of CMT.
The neck MRI findings of 33 subjects who underwent surgical release for CMT were compared with those of 18 subjects who were successfully managed only with conservative management. The MRI findings were correlated with the histopathologic sections of the CMT mass.
All 33 subjects (100%) who underwent surgical release showed one or more low signal intensities within the involved sternocleidomastoid muscle (SCM) on the T1- and T2-weighted images of neck MRI. The eighteen non-surgical candidates showed only enlargement of the SCM without low signal intensity within the SCM. The histopathologic findings showed interstitial fibrosis and/or the presence of aberrant tendon-like excessive dense connective tissue that was either well-arranged or disorganized.
The histopathologic findings and MRI findings showed good correlation in terms of the amount of fibrosis and aberrant dense connective tissue within the SCM. If multiple or large low signal intensities within the SCM are noted, we think that surgical release should be considered.
Citations
To compare the clinical severity of congenital muscular torticollis (CMT) based on the method of child birth.
Children diagnosed with CMT and who were < 6-years-of-age at the time of their first visit at the Center for Torticollis, Ajou Medical Center, were included in this study. The medical records were retrospectively reviewed with reference to the method of child birth and the clinical severity of CMT. The clinical severity of CMT was determined either by whether stretching exercises were needed for the children <6-month-of-age or whether surgical release was required for the children ≥6-months-of-age at the time of the first visit.
One hundred seventy eight subjects with CMT were enrolled. There was no significant difference in the rate of surgical release according to the method of child birth. For 132 patients <6-month-of-age there was also no significant difference in the rate of stretching exercises.
There was no significant difference in the clinical severity of CMT based on the method of child birth. This finding suggests that prenatal factors alone could be a cause of CMT and that the clinical severity of CMT in children delivered by Cesarean section is not different when compared with the severity of CMT in children born through vaginal delivery.
Citations
To investigate the coexistence rate and related factors of developmental dysplasia of the hip (DDH) and congenital muscular torticollis (CMT), and to determine whether ultrasonography (US) gives good value for screening of DDH in CMT.
We prospectively examined 121 infants (73 males and 48 females) diagnosed with CMT to determine the incidence of DDH by US. We also assessed the relationship between neck US findings and DDH occurrence, and investigated the clinical features of CMT related to DDH.
18 patients (14.9%) were diagnosed as having DDH by US. However, most DDH was subclinical and spontaneously resolved. Only 2 patients (1.7%) needed to be treated with a harness. The positive predictive value of clinical examinations for DDH was 52.6% and patients treated by harness were all clinically positive. DDH was more common in the left side (13 left, 4 right, 1 both), but 6 out of 18 DDH (33.3%) cases presented on the contralateral side of CMT. Sex difference was not observed. Breech presentation and oligohydramnios were not related to DDH occurrence. Neck US findings did not correlate with DDH occurrence.
The coexistence rate of CMT and DDH was concluded to be 14.9%. If only DDH cases that required treatment were included, the coexistence rate of these two disorders would be lowered to 1.7%. All of these patients showed positive findings in clinical examination. Therefore, hip US should not be recommended routinely for patients with CMT.
Citations
To examine whether the thickness of the sternocleidomastoid muscle (SCM) could be used as a prognostic factor for congenital muscular torticollis (CMT).
This was a retrospective study conducted in a pediatric rehabilitation service at a tertiary medical center. Fifty-two children who met the following inclusion criteria were included: 1) children who were 3 month-old or younger, 2) children diagnosed with CMT, 3) passive rotation of the face toward the shoulder of the tilted side ≤60°, 4) children who had been managed according to the clinical pathway for CMT, 5) children who had been followed up for 6 months or more after the end of treatment. The duration and total number of stretching exercise sessions were reviewed with reference to the thickness of the SCM.
Among the 52 children with CMT, 46 children were successfully managed with only stretching exercise of the SCM for 1-6 weeks (group 1: 88.5%) and 6 children were managed with botulinum toxin A injection, surgical release or both in addition to stretching exercise (group 2: 11.5%). The difference in the SCM thickness between the affected and normal sides was significantly greater in group 2 than that in group 1 (p=0.026). A strong correlation was found between the total duration of stretching exercise and the difference in the SCM thickness in group 1 (Pearson' γ=0.429; p=0.003).
Children with a thicker SCM seem to require a longer duration of stretching exercise and other therapeutic interventions in addition to stretching exercise for CMT. Therefore, the thickness of the SCM may be one prognostic factor for CMT treatment.
Citations
Method112 patients diagnosed as CMT were enrolled and we reviewed the patients' charts and radiologic findings, retrospectively. We reviewed the sex, gestational age, birth weight, mode of delivery, age at diagnosis, mass thickness, ratio of mass thickness, mass site, plagiocephaly, clavicle fracture, the frequency of rehabilitation treatment.
ResultsThe patients with a plagiocephaly or a clavicle fracture had been needed significantly longer rehabilitation and ratio of mass thickness and rehabilitative duration had a positive linear relationship and diagnostic time and the duration of rehabilitative treatment showed a positive correlation. However, rehabilitation frequency did not equate to a shorter rehabilitation period and mass site did not correlate with the duration of rehabilitation treatment. Also, the group treated with manipulation with additional ultrasound treatment showed no significant difference to the group treated with only manipulation. In this study, 2 patients received surgical treatment, which was SCM tenotomy of the affected side in conjunction with rehabilitation therapy.
ConclusionThis study showed that plagiocephaly, clavicle fracture, mass ratio, and diagnosis time are clinically significant in determining rehabilitative treatment. So, it is imperative to make a timely diagnosis and objectively evaluate the tilting of the head and neck, as well as checking the mass ratio and identifying the presence of clavicle fractures.
Objective: The ultrasonographic (US) findings of the sternoocleidomastoid muscles (SCM) in patients with congenital torticollis were studied to evaluate the correlation between the US findings and outcome of physical theraphy.
Method: We studied 52 patients diagnosed as congenital muscular torticollis. We evaluated the thickness, mass, echogenicity and echotexture of both SCM muscles. The US findings of the SCM muscles were divided into 5 grades according to the severity. The physical theraphy included heat theraphy and passive stretching of involved muscle. The outcomes of the treatment were evaluated by physical examination and follow-up US examination.
Results: Neck rotation of lesion site comparing to non-lesion site was improved significantly from 83.0⁑16.5% to 94.9⁑7.16% and lateral flexion from 77.9⁑18.1% to 90.5⁑12.4% after the treatment. In 20 US follow-up cases, thickness ratio of the involved SCM muscle decreased significantly from 177.8⁑46.2% to 158.3⁑83.1%. The therapeutic effect tends to be low with higher grades of the US findings. But, there were no significant statistical correlation between age and treatment outcome.
Conclusion: The US findings of the SCM muscles had a significant correlation with the outcome and the duration of treatment.
Objective: To evaluate the clinical usefulness of ultrasonographic measurement of the sternocleidomastoid muscle(SCM) in congenital muscular torticollis.
Method: We studied nine patients(5 males, 4 females) who were diagnosed as a congenital muscular torticollis. We measured the thickness of SCM muscles bilaterally by the ultrasonography and obstetric caliper under sedation, before and after conservative treatment.
Results: Before the treatment, thickness of the SCM muscle was significantly thicker in the lesion side than non-lesion side(p＜0.05). There was significant changes in thickness of the lesion side from the values of the before- to thoses of after-treatment(p＜0.05). Both the difference of thickness and the ratio between lesion and non-lesion side significantly decreased after the treatment(p＜0.05). Obstetric caliper measurement showed no significant changes in the difference of thickness and the ratio between the lesion and non-lesion sides after the treatment.
Conclusion: Ultrasonographic measurement of the thickness of SCM in congenital muscular torticollis was easily applicable, objective, and reliable, thus was an affordable method for both diagnosis and evaluation of the treatment effect.
The primary treatment goal of torticollis is to prevent the craniofacial deformities, limitation of neck movements and long-term postural changes.
In order to offer precise guidelines for the treatment of torticollis regarding the cosmetic and functional results, we analyzed 99 torticollis children retrospectively for the ages at diagnosis and the start of treatment, the duration of treatment, the problems at first visitation, and the size of sternocleidomastoid mass.
These patients visited the department of rehabilitation medicine, Holy Family Hospital from April 1991 to June 1997.
The results showed that there was no relation between the number of problems at first visitation and the age at the start of treatment, but the group with 1 problem had a significantly shorter duration of treatment than the groups with more than 4 problems.
The ages at diagnosis and at the start of treatment for the patients with a palpable neck mass were younger than those of the patients without a neck mass. The duration of treatment was longer for the patients with a larger size of mass by the ultrasonography of the neck.
The ages at diagnosis and at the start of treatment, and the duration of treatment were all younger and shorter in the cosmetically satisfactory group than those of the cosmeticlly unsatisfactory group. However there was no significant difference between the functionally satisfactory group and unsatisfactory group in the ages at diagnosis and at the start of treatment, and the duration of treatment.
In conclusion, when the treatment of torticollis was started early, especially at less than 3 months old, better cosmetic results were obtained, even though functional results were relatively satisfactory by the proper rehabilitation treatments. We want to emphasize that it is important to detect and treat the torticollis as early as possible for the better cosmetic results.
The clinical findings and the ultrasonographic appearance of the sternocleidomastoid muscles of 40 children with torticollis were studied to evaluate the correlation between the clinical findings and the ultrasonographic findings and to know whether the ultrasonographic findings could be the prognostic factors of the treatment outcome. This is preliminary study to find out the ultrasonographic findings of congenital muscular torticollis.
In physical examination, we checked range of motion of neck, facial asymmetry and plagiocephaly. Ultrasonsography was done in a week after the first visit and in ultrasonography, the echogenicity, echotexture, margin, size and shape of the mass were evaluated. Of 40 children, 29 children were due to congenital muscular toricollis, 9 children were due to posture(postural torticollis), and 2 children were due to strabismus. Of 29 congential muscular torticollis patients, 25 children showed fibromatosis colli(nodular form in 11 children, fusiform in 14 children) and 4 children showed only asymmetric thickness of sternocleidemast oid muscle. The relationship of the clinical findings and ultrasonographic findings were evaluated. The margin in ultrasonography and the range of motion of the neck had significant correlation(P<0.05) but the other clinical findings and other parameters of ultrasonography did not have significant correlation.
In conclusion, ultrasonography is simple method to evaluate the patients with torticollis and some of the ultrasonographic parameters have some correlation with clinical features of torticollis. To be used as a prognostic factor, further study would be necessary.
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