Congenital muscular torticollis (CMT) is a common musculoskeletal abnormality in children, which has been characterized by unclarified pathological changes in the sternocleidomastoid muscle (SCM) and various hypothetical etiologies. There are 2 main hypothetical etiologies for CMT in the literature: 1 infers that CMT may represent the sequela of an intrauterine or perinatal compartment syndrome, and the other regard CMT as a maldevelopment of the fetal SCM.To better understand the etiopathogenesis of CMT, we screened the necks of 1-day-old newborns that may potentially have CMT for evidence of SCM trauma or tumor.A convenience sample of 2564 full-term (>37 weeks) Chinese neonates were included in this study. All neonates were screened for CMT by physical examination at birth. If CMT was suspected, further ultrasonic and physical examinations were performed. When CMT was confirmed, we provided appropriate interventions and follow-up. The progress and changes in patients with CMT were recorded.Following physical examination, 44 of 2564 neonates were diagnosed with suspected CMT based on obvious facial asymmetry or palpable swelling or mass in the SCM. Among these, ultrasound examination showed 81.8% (36/44) had asymmetry in the thickness of the bilateral SCM. The 36 neonates were followed-up for 6 months; among them, 1 infant developed CMT and 35 showed normal development in bilateral SCM. The 1 patient with CMT underwent regular physiotherapy and recovered with no evidence of recurrence after the final 3 years of follow-up. No neonates suffered from signs of neck trauma, such as hematoma or subcutaneous ecchymosis.There was no evidence of neck trauma in this 1 day old newborn. The pseudotumor of SCM that developed after birth underwent differentiation, maturation, and disappeared as the baby grew. The SCM asymmetry did exist in some of the newborn babies, and became symmetric with the baby's growth. Data from this clinical study and our previous ultra-structural pathological studies suggested that both prenatal and postnatal factors play important roles in CMT. We hypothesized that CMT might be a developmental disease.
Objective:
Botulinum toxin have been used to treat congenital muscular torticollis for the last 25 years; however, few studies have been published with only limited cases and short-term follow-up. The aim of the present study is to systematically review the effectiveness and safety of botulinum toxin injections for congenital muscular torticollis by analyzing these relevant literatures.
Methods:
The authors searched PubMed, Web of Science, EMBASE, Cochrane Library, China Biology Medicine, for all articles about botulinum toxin injections for the congenital muscular torticollis. The MINORS evaluation tool was adopted to evaluate the quality of these studies. Meta-analysis calculations are made by R software 3.6.2.
Results:
This study search involved strict inclusion criteria and targeted data collection. Ten studies were included, with a total of 411 patients, comprising 1 non-randomized experimental study and 9 cases or case series. The results of our meta-analysis of single rate showed that the overall effective rate of botulinum toxin for congenital muscular torticollis was 84% (95% confidence interval [CI] 67%–96%). After botulinum toxin treatment, the conversion rate to surgery was 9% (95% CI 4%–22%), and the adverse reaction rate was 1% (95% CI 0%–3%). The most common adverse reactions among these included studies involve injection site erythema and transient dysphagia.
Conclusion:
Current evidence shows that botulinum toxin injections for the treatment of congenital muscular torticollis is safe and effective, with few serious adverse reactions. Further well-designed, larger randomized trials are warranted.
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