Congenital muscular torticollis (CMT) is a postural, musculoskeletal deformity evident at or shortly after birth. CMT results from unilateral shortening or stiffness of the sternocleidomastoid muscle (SCM) and presents as lateral flexion of the head to the ipsilateral side with rotation to the contralateral side. The incidence of CMT ranges from 0.3%1 to 16%2 of newborns. It is the third most common congenital musculoskeletal condition in newborns.3
Extensive evidence supports that physical therapy (PT) intervention is effective in resolving CMT when initiated in early infancy. However, if initiated later, CMT resolution decreases and treatment duration increases.4 When PT is initiated before 1 month of age, the prognosis for typical cervical range of motion (ROM) is 98% with 1½ months of PT.4 However, when initiated from 1 to 3 months, the prognosis for typical cervical ROM decreases to 89% with 6 months of PT; when initiated from 3 to 6 months, it decreases to 62% with 7 months of PT; and when initiated over 6 months of age, it decreases to less than 20% with 10 months of PT.4 Therefore, it is imperative that infants with CMT are identified early and receive appropriate PT intervention for optimal outcomes.
Physical Therapy Management of Congenital Muscular Torticollis: An Evidence-Based Clinical Practice Guideline from the Academy of Pediatric Physical Therapy of the American Physical Therapy Association was published in 2013 (2013 CMT CPG).5 The 2013 CMT CPG informed referral, screening, examination and evaluation, diagnosis, prognosis, intervention, consultation, discharge, and follow-up of infants with CMT. Recommendations were summarized into 16 action statements based on critical appraisal of the literature and expert opinion. The guideline development group and best practice indicate that clinical practice guidelines be updated every 5 years as evidence becomes available.6
The purpose of this study is to systematically review the recent evidence on PT diagnosis, prognosis, and intervention of CMT to inform the update to the PT management of CMT evidence-based clinical practice guideline.
This systematic review was conducted according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA).7 The search strategy for this update of the 2013 CMT CPG was similar to the original search to ensure consistency. A comprehensive search of 7 databases (CINAHL, Clinical Trials, Cochrane Library, Embase, PsycInfo, PubMed, and Web of Science) was conducted from January 2012 to September 2017 by a clinical services librarian. Medical subject headings (MeSH) and non-MeSH search terms were used, including torticollis, physical therapy, diagnosis, prognosis, and intervention. Refer to Supplemental Digital Content 1 (available at: http://links.lww.com/PPT/A214) for full search strings by database. No filters were applied for study type or language. Additional studies were identified through a manual search of the references of included studies.
Studies meeting these criteria were included: (1) participants included infants and children diagnosed with CMT, and (2) informed the diagnosis, intervention, or prognosis of CMT as related to PT. Studies were excluded based on the following criteria: (1) only on plagiocephaly, (2) dissertations and abstracts, (3) not published in English, and (4) included in the 2013 CMT CPG.5
Studies were included based on title or abstract using the inclusion and exclusion criteria. If necessary, a full-text review of the article was completed. Two authors coded the first 10% of studies (n = 206) to establish reliability for study selection. Discrepancies were resolved through discussion. The 2 authors then independently reviewed the remaining articles (n = 1856).
Study validity was appraised using the Quality in Prognostic Studies tool8 for prognostic studies, or the American Physical Therapy Association's Critical Appraisal Tool for Experimental Intervention Studies, and the Cochrane Risk of Bias9 for intervention studies. Eight reviewers completed appraisals of 3 articles to establish interrater reliability. Reviewers with 90% or more agreement were randomly assigned to appraise the remaining studies. Two reviewers appraised each study independently, scores were compared for agreement, and discrepancies were resolved via discussion. Details of the risk of bias for prognostic studies are included in Figure 1 and for intervention studies in Figures 2 and 3. In addition, intervention study designs were appraised and assigned a level of design rigor (level I, most rigorous, to level V, least rigorous) according to criteria from the American Academy of Cerebral Palsy and Developmental Medicine Systematic Review Methodology.10
Mutual consensus was used to determine the applicable data to be extracted from each study by 2 reviewers. Data extracted for prognostic studies included study design, purpose, inclusion/exclusion criteria, number of participants, timing of measures, intervention, independent variables, dependent variables, statistics, results, and clinical implications. Data extracted for intervention studies included study design, purpose, inclusion/exclusion criteria, number of participants, intervention, outcome measures, timing of measures, results for each outcome measure, and clinical implications. Data were independently extracted by 2 reviewers. Extracted data were compared for agreement, and discrepancies were resolved via discussion.
The results of the search are shown in Figure 4. The search identified 2064 studies, with 1760 studies remaining after duplicates were removed. An additional 1738 studies were excluded based on title or abstract. Twenty-two articles underwent full-text review, with 20 included in the systematic review. From the final list of included studies, 0 study informed diagnosis, 14 studies informed prognosis, and 6 studies informed intervention. Details of prognostic studies that address treatment duration are given in Table 1 and details of intervention clinical trials are provided in Table 2.
Fourteen studies informed prognosis related to the presence of an SCM lesion, extent of symptom resolution, treatment duration, adherence to PT intervention, cervical spine outcome, and motor outcome.
Presence of the SCM Lesion. Han et al11 compared clinical characteristics of infants with CMT with and without an SCM lesion on ultrasonography. Infants with an SCM lesion, compared with infants without an SCM lesion, demonstrated a younger age of presentation for medical care, greater limitations in cervical rotation and lateral flexion ROM, a greater number of breech presentations, lower proportion of cesarean deliveries, and a decreased prevalence of plagiocephaly.11
Extent of Symptom Resolution. Ryu et al12 investigated the relationship between clinical factors and symptom resolution, as quantified by no visible SCM lesion on follow-up ultrasonography. PT intervention was the only factor that correlated with complete resolution of the SCM lesion in infants with CMT.12
Lee et al13 investigated the relationship between age at initiation of treatment and symptom resolution, as quantified by postintervention head tilt, thickness ratio of the involved/uninvolved SCM, and torticollis overall assessment (TOA) scores. The TOA combines rotation and lateral flexion ROM, craniofacial asymmetry, SCM muscle quality, head tilt, and subjective assessments by parents into a single scaled score. In this study, infants with CMT younger than 6 months were separated into 2 groups based on age at initiation of treatment: before 6 weeks and after 6 weeks. Infants who initiated treatment before 6 weeks, compared with those after 6 weeks, demonstrated decreased thickness ratio of the involved/uninvolved SCM postintervention.13 There were no between-group differences for head tilt and TOA scores postintervention.13
Lee et al14 investigated the relationship between severity of preintervention neck rotation asymmetry and symptom resolution, as quantified by mid- and postintervention head tilt and TOA scores. Infants with CMT were separated into 3 groups based on severity of preintervention neck rotation asymmetry: less than 15°, 15° to 30°, and more than 30° difference between passive cervical rotation ROM of right and left sides. All groups received PT intervention for 6 months. Between-group differences for TOA scores were observed both mid- and postintervention among all groups; increased severity of initial neck rotation asymmetry was associated with decreased TOA score (lower scores reflect poorer outcomes).14 There were no between-group differences for head tilt mid- and postintervention.14
Park et al15 investigated the association between SCM muscle thickness and postintervention neck passive range of motion (PROM) assessed using the modified Cheng score, which combined neck rotation and lateral flexion PROM into a single scaled score. The involved SCM thickness preintervention, involved/uninvolved SCM thickness ratio preintervention, change in involved SCM thickness pre- to postintervention, and change in involved/uninvolved SCM thickness ratio pre- to postintervention were each moderately correlated with the modified Cheng score.15 The involved/uninvolved SCM thickness ratio preintervention demonstrated the highest correlation with the postintervention modified Cheng score, supporting that this measure may be more accurate than the preintervention involved SCM thickness alone to predict the extent of neck PROM resolution.15
Treatment Duration/Episode of Care. Six studies reported factors associated with treatment duration.11 , 16–20 A total of 777 infants participated in the studies, with sample sizes ranging from 5316 to 18211 participants. All study participants were infants with CMT younger than 1 year. For 5 studies,11 , 16–19 intervention consisted of manual stretching, strengthening, massage, positioning, therapeutic ultrasound, and/or active range of motion (AROM) activities provided 2 to 3 times per week. One study did not provide details on the intervention.20
Longer treatment duration was correlated with each of the following preintervention factors: lower birth weight,19 younger age at diagnosis,19 increased stiffness of the involved SCM,16 increased thickness of the involved SCM,19 increased thickness ratio of the involved/uninvolved SCM,18 , 19 increased degree of head tilt,18 and lower scores on the TOA.18 In addition, longer treatment duration was reported in the following groups of infants: infants with breech, compared with cephalic, presentation19; infants with motor asymmetry, compared with without motor asymmetry20; and infants with an SCM lesion present on ultrasonography, compared with infants without an SCM lesion.11
Treatment duration was significantly shorter in a group of infants with CMT with normal SCM findings on ultrasonography, compared with a group with abnormal SCM findings, when adjusting for the following preintervention factors: neck rotation PROM, neck lateral flexion PROM, and age at diagnosis.17
Adherence to Intervention. Rabino et al21 investigated factors associated with adherence to PT intervention for mothers of infants with CMT. Adherence was quantified as the proportion of prescribed exercises performed, the proportion of visits attended, and whether treatment was terminated by the clinician or prematurely by the parents. Adherence was associated with maternal perceptions of the effect of CMT on the infant's activities and the intervention's importance for the infant's future function.21 Adherence was not related to the mother's level of communication with the therapist, trust in the therapist, belief in the program, or preference of whether or not to be involved in the treatment.21
Cervical Spine Outcome. Öhman22 assessed 58 children with a history of CMT at preschool age. Only 7% exhibited a head tilt, but 26% had some degree of asymmetry in PROM.22 The clinical significance of the asymmetric neck PROM is questionable since all children had at least 85° of rotation to each side, and the 7 children with a lateral flexion difference had a difference between sides of only 5° to10°.22 Asymmetric neck PROM at preschool age was associated with the degree of asymmetric neck rotation PROM as an infant.22
Motor Outcome. Three studies informed the association of CMT with gross motor outcome.20 , 23 , 24 In a retrospective analysis, Watemberg et al20 found that, at initial evaluation, delayed motor development was significantly more common in infants with postural CMT with functional asymmetry, versus without asymmetry.20
Cabrera-Martos et al24 investigated the acquisition of 4 gross motor skills (rolling, sitting, crawling, and standing without support) for 3 groups of infants: infants with deformational plagiocephaly (DP) and acquired CMT, infants with DP and congenital CMT, and infants with DP alone. When controlling for age at referral and severity of DP, all groups demonstrated similar age at initiation of rolling and sitting.24 Infants with DP and CMT (acquired and congenital) crawled and stood without support earlier than infants with DP alone.24
Ohman and Beckung23 investigated the association of CMT with gross motor development at preschool age. History of CMT during infancy was not associated with gross or fine motor delays at 3.5 to 5 years, as assessed using the Movement Assessment Battery for Children, second edition (MABC-2).23
Five studies informed intervention, including neck PROM, microcurrent, kinesiology taping, group therapy, and postsurgical intervention.
Neck Passive Range of Motion. He et al25 informed dosage of neck stretching. This randomized control trial (RCT) compared stretching 50 times a day to stretching 100 times a day. Both dosages resulted in improved head tilt, neck PROM, and decreased SCM thickness in infants with CMT.25 However, stretching 100 times a day resulted in greater improvement in head tilt and neck PROM.25
Microcurrent. Kwon and Park26 informed the use of microcurrent. This RCT compared therapeutic exercise and ultrasound with or without additional microcurrent. The group receiving microcurrent demonstrated a shorter treatment duration, improved neck rotation PROM, and decreased involved SCM thickness, cross-sectional area, and red pixel intensity.26
Kinesiology Taping. Two RCTs informed the use of kinesiology taping (KT). One RCT compared exercises with KT applied to the involved SCM for inhibition, KT applied to the involved SCM for inhibition and to the uninvolved SCM for facilitation, and no KT.27 All 3 groups demonstrated improved neck PROM, Muscle Function Scale (MFS) scores, and Plagiocephaly Severity Scale scores, with no significant differences between groups.27 One exception was that there was no improvement in neck rotation PROM in the group with the KT applied to both SCMs.27
Another RCT compared the immediate effects of KT applied to the involved SCM for inhibition with a comparison group without KT application.28 The KT group, versus the comparison group, demonstrated improved symmetry in active neck lateral flexion strength as indicated by a significantly lower MFS score on the involved side and a significantly higher MFS score on the uninvolved side.28
Group Therapy. Surprenant et al29 informed use of group therapy. This cohort study evaluated the use of a group-based, team service delivery model for caregivers of 35 infants with CMT. Results indicated significant improvements in the infants' preferred resting head position, head shape, head tilt, and facial asymmetry.29 In addition, parents exhibited significant gains in knowledge of CMT and reported that they were very satisfied with the group program, rating it excellent quality.29
Postsurgical Intervention. Oledzka and Suhr30 informed postsurgical PT intervention of CMT. This case report described intervention of two 2-year-old children, status post-SCM surgical release. The children received PT intervention for 3 and 11 months for pain management, functional mobility, ROM, strengthening, activities to promote midline, vestibular treatment, scapular stabilization, bimanual activities, proprioception exercises, Kinesiotaping, and use of TOT Collar. At the first postoperative visit with the surgeon, a soft cervical collar was discontinued and a pinless halo was initiated until 10 to 12 weeks postsurgery. Families were provided with a daily home program. Both children achieved full ROM, symmetrical cervical strength, midline head position, performed transitions with symmetrical head righting, and demonstrated age-appropriate gross motor skills.30
The findings from this systematic review inform 6 action statements in the 2013 CMT CPG.5 The discussion has been organized by each action statement.
Action Statement 3: Document Infant History
The 2013 CMT CPG5 recommends documenting 9 specific health history factors. This review provides additional support for documenting age at initial visit,13 , 17 , 19 and delivery history, including birth presentation and history of breech presentation,19 since these variables have been associated with either symptom resolution and/or treatment duration. This review adds that delivery history should include low birth weight,19 as this has been associated with a longer treatment duration.
Action Statement 9: Examine Activity and Developmental Status
The 2013 CMT CPG5 cautioned that infants with CMT may be at risk for delays in early motor development.31 Öhman et al31 found that infants with CMT had lower scores on the Alberta Infant Motor Scale32 at 2 and 6 months when compared with infants without CMT. However, they noted the risk of delay seemed to be more associated with little or no time in the prone position while awake, than with CMT.31 In addition, gross motor function was not different between groups at 10 and 18 months of age.31 In this review, Cabrera-Martos et al24 found similar age of attainment of rolling and independent sitting among infants with CMT and DP versus infants with DP alone. However, infants with CMT and DP stood and walked earlier than those with DP alone. It is important to note that the 95% confidence interval for walking in the DP-alone group was 12.8 to 14.35, which is considered within normal limits for walking attainment.
Action Statement 11: Determine Prognosis
The 2013 CMT CPG5 states that prognoses for the extent of symptom resolution, the episode of care, and/or the need to refer for more invasive interventions are related to the age of initiation of treatment, classification of severity, intensity of intervention, presence of comorbidities, rate of change, and adherence with home programming.5
The results of this review provide additional support that PT intervention is critical for complete resolution of the muscular lesion of CMT in infants12 and that infants achieve improved symptom resolution with earlier age of initiation of treatment.13 This review provides further support for the first 3 grades of the classification of severity proposed in the 2013 CMT CPG.5 In the 2013 CMT CPG, infants younger than 6 months are classified into 3 groups based on preintervention passive neck rotation asymmetry: grade 1 (<15°), grade 2 (15°-30°), and grade 3 (>30°).5 These are the same 3 groups used by Lee et al,14 who found that increased severity of initial neck rotation asymmetry between right and left sides was associated with decreased symptom resolution as assessed using the TOA score. This review also adds that the involved/uninvolved SCM thickness ratio preintervention may be more accurate than the preintervention involved SCM thickness alone to predict the extent of neck PROM resolution.15
The results of this review confirm that the episode of care or treatment duration is related to the extent of the fibrous mass.11 , 14 , 16 , 19 Infants with an SCM lesion, compared with those without a lesion, exhibit longer treatment duration,11 and increased stiffness and thickness of the involved SCM is associated with longer treatment duration.14 , 16 , 19 The results of this review add that history of breech presentation,19 low birth weight,19 and presence of motor asymmetry20 are also associated with increased treatment duration. Contrary to the literature in the 2013 CMT CPG5 that associated shorter treatment duration with younger age of initiation of treatment,4 , 33–35 Jung et al19 found that younger age of initiation of treatment resulted in longer treatment duration. The authors of this study noted that the participants with more severe symptoms were referred for assessment earlier than those with less severe symptoms, which may explain this finding.19
Action Statement 12: Provide the Following 5 Components as the First-Choice Intervention
The 2013 CMT CPG5 states that the PT plan of care for infants with CMT or postural asymmetry should minimally address the following 5 components: neck PROM, neck and trunk AROM, development of symmetrical movement, environmental adaptations, and parent/caregiver education. The findings from this systematic review further support the recommendations for neck PROM as a component of the first-choice intervention. Our results add that increased frequency of neck PROM exercises may result in improved outcomes.25 Our results also add that, to increase adherence to PT intervention, therapists should educate parents on the effect of CMT on the infant's activities and the intervention's importance for the infant's future function.21
Action Statement 13: Provide Supplemental Interventions, After Appraising Appropriateness for the Infant, to Augment the First-Choice Intervention Obtained
The 2013 CMT CPG5 states that supplemental interventions may be added when first-choice interventions have not improved ROM or postural alignment, when access to services is limited, or when the infant is unable to tolerate the intensity of the first-choice intervention. This review provides higher-level RCT evidence for the use of microcurrent in addition to PT to improve outcomes and reduce treatment duration for infants with CMT,26 but suggests that KT may not provide added benefit when added to PT intervention.27 While higher-level evidence is emerging on microcurrent and KT, further research is needed.
Action Statement 16: Provide Follow-up Screening of Infants 3 to 12 Months Postdischarge
The 2013 CMT CPG5 recommends a follow-up reassessment after discontinuation of direct PT intervention to examine positional preference, structural and movement symmetry, and developmental milestones.
This review provides support for reassessment after discontinuation of direct PT, since a residual head tilt and neck PROM asymmetries were noted in a cohort of preschool children with a history of CMT.22 Specifically, infants with greater neck rotation PROM asymmetries may be at greater risk for neck PROM asymmetries at preschool age.22
This review also supports that a history of CMT was not associated with gross or fine motor delays at 3.5 to 5 years, as assessed by the MABC-2.23 However, a study by Schertz et al36 found that a cohort of 7- to 9-year-old children with a history of CMT had a greater prevalence of attention-deficit hyperactivity disorder and developmental coordination disorder than would be expected in the general population. Type of CMT (SCM mass or tightness vs postural CMT) did not predict risk for a neurodevelopmental disorder, but children with a history of postural torticollis had significantly lower mean scores on the MABC.36 Although more evidence is needed, a follow-up reassessment as recommended by the 2013 CMT CPG,5 is warranted to assess for asymmetries or developmental delays.
Incomplete retrieval of references through database searching constituted a limitation at the review level. Manual search via cross-referencing relevant studies was undertaken to address this limitation.
This review provides new evidence supporting that low birth weight,19 breech presentation,19 and presence of motor asymmetry20 are prognostic factors associated with longer treatment duration. New evidence also supports that increased stretching frequency results in improved outcomes for infants with CMT.25 Higher-level evidence is emerging on microcurrent26; however, further evidence is needed to establish its efficacy.
We are grateful to the following individuals who assisted with the critical appraisals: Jennifer Donenberg, PT, DPT, PCS; Alina Marrone, PT, DPT; Bianca Mendonca, PT, DPT, PCS; Susan Knight, PT, PCS; Sara Peterson, PT, DPT, PCS; and Jeremy Wong PT, DPT, PCS. We are also grateful for the feedback provided by Sandra Kaplan, PT, DPT, PhD, and Colleen Coulter, PT, DPT, PhD, PCS, who reviewed the manuscript before submission.
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