INTRODUCTION
Torticollis is one of the most common deformities seen in infants, usually presenting with rotation and lateral flexion of the head and neck.1 It is generally unilateral, but may be bilateral in rare cases.2
There are 2 main types of congenital torticollis. In postural torticollis, the infant has a postural preference but no restriction of passive range of motion. Muscular torticollis involves fibrosis and contracture of the sternocleidomastoideus muscle (SCM) on one side, causing lateral flexion of the neck to the same side and rotation to the opposite side.3 The accumulation of collagen and fibroblasts around the muscle fibers leads to atrophy and asymmetric muscle strength between the right and left sides. Over time, thickening and stiffness of the SCM results in restricted rotation and lateral flexion of the neck.4 These restrictions can hinder head movement, resulting in positional plagiocephaly.5 Moreover, infants with torticollis exhibit transient motor asymmetry and delayed motor milestones.6,7 Musculoskeletal disorders such as foot anomalies, brachial plexus lesion, and hip dislocation may also coexist with torticollis.8–10
Although the etiology of torticollis remains uncertain, possible causes include trauma during delivery, intrauterine malposition and ischemia during the third trimester, decreased amniotic fluid volume, and in utero compartment syndrome.11–13
General movements (GMs) are age-specific movements of the entire body and are characterized by complexity, fluency, and variation in the speed, frequency, and direction.14 At approximately 5 months post-term, GMs are gradually replaced by voluntary goal-oriented movements. GM assessment is a valid and reliable method of detecting early brain damage and dysfunction.14 Based on visual Gestalt perception, the character of GMs is differentiated into 3 periods: preterm movement period, writhing movement period, and fidgety movement period.15 Fidgety movements, which are seen between 3 and 5 months of age, are circular movements in all directions that have small amplitude and moderate speed.16 These movements are believed to have an adaptive function in calibrating the proprioceptive system.17 Neurodevelopmental outcomes can best be predicted by assessment of fidgety movements at 3 to 5 months.17 A detailed assessment of posture and motor repertoire increases the predictive value for development.18 The assessment yields a Motor Optimality Score (MOS) that reflects the overall quality of motor repertoire. Assessing of the early motor repertoire can help identify infants who may be at risk of lower gross and fine motor performance.19 In addition, poor early motor repertoire has been associated with minor neurological dysfunction20 and lower intelligence level21 at school ages.
Torticollis may be accompanied by developmental motor delay7 and other neurological or musculoskeletal conditions.22 Evaluating the motor repertoire of infants with torticollis may suggest neurodevelopmental problems in the early months of life and provide information about the functional integrity of the brain. This investigated the following questions: (i) Do the movements and postures of 3- to 5-month-old infants with torticollis differ compared with a control group composed of infants developing typically without torticollis? (ii) Are the clinical characteristics of 3- to 5-month-old infants with torticollis associated with their MOS?
METHODS
Participants were infants 3 to 5 months corrected age, referred to our clinic between 2019 and 2020, and diagnosed with postural or muscular torticollis. Infants with other causes of torticollis (ocular, neurological, and structural, Klippel Feil), epilepsy, or brain injury were excluded from the study. The control group comprised infants who were matched to the group with torticollis in terms of gestational age and video recording age, were followed until at least 2 years of age, and had typical development. In total, 40 infants with torticollis (21 girls, 19 boys) and 40 infants developing typically without torticollis (17 girls, 23 boys) participated.
Infants with torticollis were classified according to “Congenital Muscular Torticollis Classification Grades and Decision Tree for 0-12 months—2018 Update” (grades 1 to 8) based on the infant's age at examination, the presence of an SCM mass, and the difference in cervical rotation passive range of motion between the left and right sides.8
Clinical characteristics including gestational age, birth weight, sex, cesarean delivery, presence of plagiocephaly, side of torticollis, and developmental dysplasia of the hip were collected from the infants' medical records (Table 1).
TABLE 1 -
Clinical Characteristics of the Infants
|
Torticollis (n = 40) |
Control Group (n = 40) |
| Gestational age, mean ± SD, wk |
39.4 ± 0.9 |
39.0 ± 0.9 |
| Birth weight, mean ± SD, g |
3393.2 ± 394.2 |
3226.1 ± 422.1 |
| Sex, female, n (%) |
21 (52.5) |
17 (42.5) |
| Cesarean delivery, n (%) |
10 (31) |
13 (40.6) |
| Plagiocephaly, n (%) |
15 (37.5) |
3 (7.5) |
| Side of torticollis, right, n (%) |
26 (65.0) |
... |
| Developmental dysplasia of the hip, n (%) |
2 (5) |
... |
| Classification of torticollis |
Grade 1 |
Grade 2 |
Grade 3 |
... |
|
13 |
15 |
12 |
|
Between 9 and 15 weeks post-term, a single 3- to 5-minute video was recorded for each infant during a period of active wakefulness while in a supine position. Detailed GM assessment was performed according to the 2019 revised form.18 This assessment comprises 5 subcategories: fidgety movements, observed movement patterns, age-adequate movement repertoire, observed postural patterns, and movement character. These subscale scores are summed to obtain the MOS, which ranges from 5 to 28 points. An MOS of 25 or more is considered optimal, while an MOS of 14 or less indicates the infants should receive early intervention.18
GM assessment of all infants was performed retrospectively by 2 raters, one of whom was blind to the infants' groups. In case of disagreement, the raters reevaluated the records until a consensus was reached.
The Non-Interventional Clinical Research Ethics Board approved the research plan (2012-KAEK-15/1986). All families signed a declaration of informed consent.
Statistical Analysis
SPSS for Windows 23 software was used for statistical analyses. Visual and analytical methods were used to evaluate whether the data were normally distributed. Descriptive statistics for numerical data were presented as mean, standard deviation, median, and interquartile range. Number and percentage values were calculated for the categorical data. The Mann-Whitney U test was used for comparison of 2 independent groups if at least 1 data set had nonnormal distribution. The Kruskal-Wallis test was used to compare 3 nonnormally distributed groups. The Pearson χ2 test or Fisher exact test was used for the analysis of categorical data. Spearman correlation analysis was used to evaluate correlations between variables if at least 1 did not have a normal distribution or was ordinal. Intraclass correlation coefficients were calculated to test interobserver agreement for the MOS and subcategory scores. The statistical significance level was P < .05.
RESULTS
The study and control groups had similar scores for fidgety movements, observed movement patterns, and movement character. However, the group with torticollis had the significantly lower MOS (P < .001) and subcategory scores for age-adequate movement repertoire (P = .029) and observed postural patterns (P = .004) compared with the control group (Table 2).
TABLE 2 -
Comparison of Motor Optimality Score and Subcategory Scores of 2 Groups
|
Torticollis (n = 40) |
Control Group (n = 40) |
P Value |
|
Median (25%-75% IQR) |
Median (25%-75% IQR) |
| Motor Optimality Score |
24 (22.5-26.0) |
26 (24-27) |
<.001a
|
|
n (%)
|
n (%)
|
|
| Fidgety movement score |
|
|
... |
| Absent |
... |
... |
|
| Abnormal |
... |
... |
|
| Normal |
40 (100) |
40 (100) |
|
| Observed movement pattern score |
|
|
... |
| N < A |
... |
... |
|
| N = A |
... |
... |
|
| N > A |
40 (100) |
40 (100) |
|
| Age-adequate movement repertoire score |
|
|
.029b
|
| Absent |
5 (12.5) |
1 (2.5) |
|
| Reduced |
17 (42.5) |
10 (25.0) |
|
| Present |
18 (45.0) |
29 (72.5) |
|
| Observed postural pattern score |
|
|
.004b
|
| N ˂ A |
7 (17.5) |
3 (7.5) |
|
| N = A |
14 (35.0) |
4 (10.0) |
|
| N > A |
19 (47.5) |
33 (82.5) |
|
| Movement character score |
|
|
.189b
|
| CS |
... |
... |
|
| Abnormal, not CS |
33 (82.5) |
28 (70) |
|
| Smooth and fluent |
7 (17.5) |
12 (30) |
|
Abbreviations: A, abnormal; CS, cramped synchronized; IQR, interquartile range; N, normal.
aMann-Whitney U test.
bPearson χ2 test.
All infants in the group with torticollis and control group had normal fidgety movements and predominantly normal observed movement patterns. Age-inadequate movement repertoire was observed in 5 infants in the group with torticollis and 1 infant in the control group. In addition, postural patterns were more atypical than typical in 19 infants in the group with torticollis and 3 infants in the control group, and movement character was not fluent and smooth in 33 infants in the group with torticollis and in 28 infants in the control group (Table 2).
There were no statistically significant differences in the MOS according to torticollis grade (P = .57) or other clinical features of the infants in the group with torticollis (Table 3).
TABLE 3 -
Analysis of Clinical Features and Motor Optimality Scores of Infants
|
Torticollis (MOS) (P Value) |
Control Group (MOS) (P Value) |
| Gestational agea
|
0.55 |
0.58 |
| Birth weighta
|
0.12 |
0.43 |
| Sexa
|
0.64 |
0.96 |
| Cesarean deliverya
|
0.89 |
0.88 |
| Plagiocephalya
|
0.45 |
0.13 |
| Side of torticollisa
|
0.31 |
... |
| Classification gradesb
|
0.57 |
... |
Abbreviation: MOS, Motor Optimality Score.
aSpearman correlation coefficient.
bKruskal-Wallis test.
Interobserver agreement was excellent for the MOS and its subcategories (Table 4).
TABLE 4 -
Interrater Agreement for Motor Optimality Scores and Subcategory of All Infants
a
|
ICC (95% Confidence Interval) |
P Value |
| Motor Optimality Score |
0.948 (0.914-0.968) |
<.001 |
| Fidgety movements |
0.907 (0.847-0.944) |
<.001 |
| Observed movement patterns |
0.935 (0.877-0.966) |
<.001 |
| Age adequate movement repertoire |
0.941 (0.904-0.964) |
<.001 |
| Observed postural patterns |
0.948 (0.914-0.968) |
<.001 |
| Movement character |
0.898 (0.833-0.938) |
<.001 |
Abbreviation: ICC, intraclass correlation coefficient.
aICC, interscorer agreement; P < .05 (statistically significant).
DISCUSSION
This study aimed to evaluate the movements and postures of infants with torticollis at 3 to 5 months of age compared with infants developing typically without torticollis at the same age. Although infants with and without torticollis had similar scores for fidgety movements, observed movement patterns, and movement character, those with torticollis had the lower MOS and subcategory scores for age-adequate movement repertoire and observed postural patterns.
Many studies have examined detailed GM analysis of motor repertoire in infants with different risks factors and identified distinct movement patterns associated with these risks. Infants with Down syndrome had the lower MOS in the fidgety period than infants without Down syndrome, as well as heterogeneity of fidgety movements, lack of movements to the midline, and several atypical postures.23 In another study, it was determined that the motor repertoire of infants with cystic fibrosis was age-inadequate in the fidgety period, and the MOS may facilitate the prediction of disease severity in cystic fibrosis.24 Alkan et al25 evaluated motor repertoire during the fidgety period in infants with hypoxic-ischemic encephalopathy and determined that they had poorer motor repertoire than peers and that motor repertoire was negatively associated with the severity of hypoxic-ischemic encephalopathy. The infants included in these studies had central nervous system problems or genetic disorders. Therefore, it is expected that these problems are reflected in and alter the motor repertoire. When motor repertoire during the fidgety period was compared between infants with and without obstetric brachial plexus lesion (OBPL), no differences were observed in the MOS or subcategory scores.26 As there is no central nervous system insult in infants with OBPL, there was no statistical difference reflected in the results of detailed GM analysis. We expected the same results for infants with torticollis. All of the infants in the group with torticollis had normal fidgety movements and the MOS within the typical range. However, there were differences in the MOS (P < .001), age-adequate movement repertoire scores (P = .029), and observed postural pattern scores (P = .004) between the 2 groups. This may be attributable to the impact of suboptimal personal and environmental factors (less visual stimulation due to head preference, less awake time in prone position, less parental cooperation with home activity program, etc) on development. Our detection of these differences at 3 to 4 months of age emphasizes the importance of optimizing these factors as early as possible.
A study researching the motor asymmetry of children with torticollis reported that these children had motor asymmetry and delay in motor development (14.5% of infants), which improved in 2 years.6 Another study stated that infants with torticollis were at increased risk of delays in early gross motor functions for 1 year, after which the rate of gross motor delay was similar to that in the general population.27 In a study of high-risk infants, the presence of fidgety movements and abnormal concurrent motor repertoire was found to be associated with impaired cognitive and motor outcomes later in life.28 Consistent with these studies, the differences in the MOS, age-adequate motor repertoire score, observed postural pattern scores, and no difference in fidgety movement scores found in this study may be indicators of delays in motor development in infants with torticollis, ages 3 to 5 months. Therefore, detailed GM analysis for the early recognition of later developmental problems in infants with torticollis may benefit infant evaluation.
In addition, our study demonstrates which aspects of the movement repertoire differ between groups. Considering these specific differences, supporting infants with torticollis in terms of promoting symmetrical body posture and age-adequate movement repertoire may optimize their motor development. It is important to encourage movements toward the midline and against gravity, such as hand-to-hand contact, foot-to-foot contact, hand-to-mouth contact, and leg lift.
Schertz et al29 reported attention-deficit hyperactivity disorder, developmental coordination disorder, language impairment, and autistic spectrum disorder as later neurodevelopmental conditions in children with torticollis. Butcher et al21 also determined that the low MOS was associated with sub-optimal cognitive function at school age. As the infants in our study had normal fidgety movements but the lower MOS than the control group, they may be at risk for the adverse outcomes listed earlier. However, long-term follow-up of all infants in the group with torticollis is necessary to verify or exclude this idea.
Herrero et al23 did not identify a relationship between the MOS or fidgety movements and the clinical features of infants with Down syndrome. Similarly, in our study, there was no correlation found between the MOS and the clinical features of infants with torticollis. A review by Martiniuk et al30 showed that plagiocephaly is a marker of elevated risk of developmental delays. In our study, 37.5% of the infants had plagiocephaly, but plagiocephaly was not associated with the MOS. Plagiocephaly is often the result of torticollis. Therefore, torticollis is the reason for both plagiocephaly and the difference in the MOS, which may explain the lack of a significant relationship between the 2 results. Our findings that the MOS did not differ according to torticollis grade may also be related to the low torticollis grades of the infants in our sample (grades 1, 2, and 3). Moreover, the numbers of infants in each grade were low (n = 13, 15, and 12, respectively).
Limitations
One of the researchers who performed the detailed GM assessment was blind to the infants' group. However, the other researcher was not blinded. Considering the possibility that this may lead to bias, we performed interobserver agreement analysis and the results indicated excellent consistency. While this suggests a lack of bias in our study, it is still not possible to prevent the observers from noticing obvious torticollis during analysis.
The lack of long-term follow-up is a limitation of the study. Further studies with longer follow-up and larger samples are needed.
CONCLUSIONS
Infants with torticollis had lower MOS and subcategory scores for age-adequate movement repertoire and observed postural patterns when compared with peers. Thus, strategies supporting the movement repertoire of infants with torticollis may be added to rehabilitation programs for infants with lower MOS, which will help optimize their motor development.
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