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Is Longstanding Congenital Muscular Torticollis Provoking Pelvic Malalignment Syndrome?

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Is Longstanding Congenital Muscular Torticollis Provoking Pelvic Malalignment Syndrome? children Article Is Longstanding Congenital Muscular Torticollis Provoking Pelvic Malalignment Syndrome? Jun-il Park, Joo-Hyun Kee , Ja Young Choi and Shin-seung Yang * Department of Rehabilitation Medicine, College of Medicine, Chungnam National University, Daejeon 35015, Korea; [email protected] (J.-i.P.); [email protected] (J.-H.K.); [email protected] (J.Y.C.) * Correspondence: [email protected] Abstract: It has been reported that congenital muscular torticollis (CMT) may result in secondary scoliosis over long-term follow-ups. However, there are few reports on whether CMT causes pelvic malalignment syndrome (PMS). This study aimed to investigate the relationship between CMT and PMS and to determine the factors associated with the development of PMS in children with longstanding CMT. Medical records of 130 children with CMT who had long-term follow-up were reviewed retrospectively. The chi-squared test and logistic regression analysis were used to determine which initial clinical parameters contributed to the development of PMS. Among 130 children with CMT, 51 (39.2%) developed PMS with or without compensatory scoliosis during long-term follow-up, indicating a high prevalence of PMS in children with a CMT history. Initial clinical symptoms such as a limited range of motion of the neck or the presence of a neck mass could not predict the development of PMS. Even if the clinical symptoms are mild, long-term follow-up of children with CMT is essential to screen for PMS. Citation: Park, J.-i.; Kee, J.-H.; Choi, Keywords: pelvic malalignment syndrome; congenital muscular torticollis; long-term follow-up; scoliosis J.Y.; Yang, S.-s. Is Longstanding Congenital Muscular Torticollis Provoking Pelvic Malalignment Syndrome? Children 2021, 8, 735. 1. Introduction https://doi.org/10.3390/ Congenital muscular torticollis (CMT) is a clinical symptom that commonly occurs children8090735 in neonates and infants [1]. It is also known as wry neck and twisted neck, and in the Academic Editor: Erich Rutz case of mass-type CMT, it is called fibromatosis colli [2–5]. Shortening of the unilateral sternocleidomastoid (SCM) muscle is characteristic of the disease, and it is known to occur Received: 7 June 2021 in 0.3–2.0% of newborns [6–8]. Contracture of the SCM muscle causes the face to rotate to Accepted: 24 August 2021 the opposite side and the head to tilt to the same side, resulting in signs and symptoms Published: 26 August 2021 such as a limited range of motion (LOM) in the neck [9]. Without appropriate treatment, long-term twisting of the neck may result in compensatory changes in the adjacent skeletal Publisher’s Note: MDPI stays neutral structures, resulting in bilateral facial asymmetry and secondary scoliosis [9–12]. with regard to jurisdictional claims in The etiology of CMT remains unclear. However, previous studies have assumed that published maps and institutional affil- congenital trauma, perinatal compartment syndrome, and impairment of the developing iations. SCM owing to intrauterine constraints are the main causes [13]. Additionally, secondary changes in the infant’s spine have been reported [14–16]. Recent studies have shown that the severity of spinal deformities in infants with CMT increases with age and with the severity of SCM tightening [13,14]. Copyright: © 2021 by the authors. Pelvic malalignment syndrome (PMS) is a biomechanical change that might cause Licensee MDPI, Basel, Switzerland. pain when the pelvis supporting the central axis of the body is twisted, and the entire body This article is an open access article is out of alignment [17]. Rotational malalignment, upslip of the sacroiliac joint, and inward distributed under the terms and or outward movement are common, and if growth continues without modification, the conditions of the Creative Commons spine and pelvis are deformed [17,18]. In severe cases, the disk may be pushed out to one Attribution (CC BY) license (https:// side, causing spinal diseases that exert pressure on the surrounding nerves. It was reported creativecommons.org/licenses/by/ that there was a positive correlation between the severity of scoliosis and pelvic obliquity 4.0/). Children 2021, 8, 735. https://doi.org/10.3390/children8090735 https://www.mdpi.com/journal/children Children 2021, 8, 735 2 of 10 in patients with adolescent idiopathic scoliosis. There were some children seen with both secondary scoliosis and pelvic asymmetry among the children with a history of CMT [19]. Although secondary scoliosis frequently occurs in patients with CMT, the relation- ship between CMT and PMS has not been evaluated. To the best of our knowledge, no study regarding the development of PMS through long-term follow-up of children with CMT exists. Therefore, our study aimed to investigate the relationship between CMT and PMS and to determine the factors associated with the development of PMS in children with longstanding CMT. 2. Materials and Methods 2.1. Study Design This retrospective study was conducted at a single tertiary medical center. We re- viewed the medical records of children who were diagnosed with CMT between January 2011 and June 2017. This study was approved by the institutional review board of the National University Hospital (IRB no. 2017-08-008-001). 2.2. Subjects We reviewed the medical records of 130 children (80 boys and 50 girls) who visited our outpatient clinic of the rehabilitation department between January 2011 and June 2017 (Table1). The inclusion criteria were children who were diagnosed with CMT for the first time before the age of 4 years and had been followed up for more than 2 years. The included children had visited the hospital with the chief complaint of abnormal posture of the head and were diagnosed with CMT by ultrasonography and clinical examination. We enrolled both mass-type and non-mass-type CMT cases. In most cases of mass type, the mass had disappeared before 12 months, and that was confirmed by clinical examination and ultrasonography during the routine follow-up examination. Babies under 1 year were routinely followed up every 2 months. The exclusion criteria were as follows: (a) congenital anomalies of the spine, (b) spasmodic torticollis, (c) premorbid or comorbid musculoskeletal problems that affect the cervical range of motion, and (d) other congenital musculoskeletal anomalies of a lower extremity, such as leg length discrepancy or hip dysplasia. In addition, children who underwent surgical treatment for the remaining fibrotic mass were excluded from this study. Table 1. Characteristics of children with CMT (n = 130). Characteristic n (%) Male 80 (61.5) Sex Female 50 (38.5) 0–12 months 124 (95.4) Initial diagnosis age 13–24 months 3 (2.3) After 24 months 3 (2.3) Cesarean section 38 (29.2) Delivery type Vaginal delivery 92 (70.8) Limited 73 (56.2) Range of motion Normal 57 (43.8) Presence 45 (34.6) Presence of fibrotic mass Absence 85 (63.4) Presence 34 (26.2) Plagiocephaly Absence 96 (73.8) Note: Values are presented as numbers (%). Children 2021, 8, x FOR PEER REVIEW 3 of 10 Note: Values are presented as numbers (%). Children 2021, 8, 735 Almost all children diagnosed with CMT received regular physical3 of therapy 10 , and their parents were educated to do stretching of affected SCM muscles daily, even if the patients did not get the regular physical therapy. Almost all children diagnosed with CMT received regular physical therapy, and their parents2.3. Outcome were educated Measurements to do stretching of affected SCM muscles daily, even if the patients did notChildren get the regular presenting physical with therapy. observable neck/head/facial asymmetry were screened for 2.3.a diagnosis Outcome Measurements of CMT. Diagnosis of congenital muscular torticollis was usually based on BallockChildren’s algorithm presenting [8]. with The observable birth history neck/head/facial was obtained asymmetry by the were caregiver screened’ fors interview. a diagnosisPhysical of CMT. examinations Diagnosis of were congenital performed muscular at torticollisthe first wasvisit usually and the based regular on 2-month-in- Ballock’sterval follow algorithm-ups [8 during]. The birth the history first year was obtainedof life and by the then caregiver’s were executed interview. annually. Physical examinations were performed at the first visit and the regular 2-month- intervalPhysical follow-ups examinations during the first for year the of neck life and included then were SCM executed tightness, annually. the presence of a fibrotic mass,Physical the presence examinations of plagiocephaly, for the neck included and SCM passive tightness, neck the range presence of motion of a fibrotic (ROM). At every mass,hospital the presencevisit, the of degree plagiocephaly, of SCM and tightness passive neck was range checked of motion through (ROM). stretching At every of each sternal hospitalbelly and visit, cleido the degree-belly of, SCMand tightnessthe severity was checked of plagiocephaly through stretching was of assessed each sternal using a diagonal bellycaliper and, which cleido-belly, measured and the the severity difference of plagiocephaly between wasthe assesseddiagonal using lengths a diagonal. Passive neck ROM caliper, which measured the difference between the diagonal lengths. Passive neck ROM waswas measured measured on on both both sides sides using using a goniometer a goniometer in the transversein the transverse and coronal and planes coronal planes (Fig- (Figureure 1)1 during) during the firstfirst year year of life.of life. Figure 1. Cervical range-of-motion measurement using arthrodial goniometer (adapted from Hwang Figure 1. Cervical range-of-motion measurement using arthrodial goniometer (adapted from et al. Changes in muscle stiffness in infants with congenital muscular torticollis 2019; 9(4); 158 [20]). Hwang et al. Changes in muscle stiffness in infants with congenital muscular torticollis 2019; 9(4); 158 [If20 there]). was a difference in the ROM of the SCM between the affected side and the unaffected side in either plane, we marked the case to have the LOM of the neck.
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