Bilateral congenital muscular torticollis in infants, report of two cases

Anna Öhman

doi:10.12688/f1000research.143499.2

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Clinical Practice Article

Revised

Bilateral congenital muscular torticollis in infants, report of two cases

[version 2; peer review: 3 approved with reservations, 1 not approved]

Anna Öhman

PUBLISHED 17 Jun 2024

Author details Author details

Health and Rehabilitation/Physiotherapy, University of Gothenburg, Gothenburg, Sweden

Anna Öhman
Roles: Conceptualization, Investigation, Writing – Original Draft Preparation

OPEN PEER REVIEW

REVIEWER STATUS

Abstract

Background

Congenital muscular torticollis (CMT) is a well-known diagnosis among physiotherapists specializing in pediatric care, especially when working with infants. However, knowledge of bilateral torticollis is limited. The purpose of this article was to describe how bilateral torticollis may present itself clinically.

Case

One infant with CMT with sternocleidomastoid tumor (SMT) on the right side, and some limitation in rotation towards the right side and in lateral flexion towards the left side, i.e, the muscle on the right side was shortened. While sitting with support, he tilted the head to the left and was stronger in the lateral flexors on the left side which fit well with a postural left-sided torticollis (PT). The other infant had bilateral muscular torticollis (MT), the sternocleidomastoid muscle thickened bilaterally, and both active and passive rotations were affected. The head was held in flexion, and active rotation was severely limited to both sides. For both cases the therapeutic interventions were to gain a normal range of motion (ROM) and a good posture of the head.

Conclusions

CMT can appear in different ways and may be bilateral. Both infants gained good ROM and better head position, however case I still needs some training. To gain more knowledge about bilateral CMT, we should follow these cases over a longer period of time. It is important to communicate and discuss our experiences with each other to understand rare cases of CMT.

Keywords

Torticollis, bilateral, infant, physiotherapy

Corresponding author: Anna Öhman

Competing interests: No competing interests were disclosed.

Grant information: The author(s) declared that no grants were involved in supporting this work.

Copyright: © 2024 Öhman A. This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

How to cite: Öhman A. Bilateral congenital muscular torticollis in infants, report of two cases [version 2; peer review: 3 approved with reservations, 1 not approved]. F1000Research 2024, 13:211 (https://doi.org/10.12688/f1000research.143499.2) First published: 21 Mar 2024, 13:211 (https://doi.org/10.12688/f1000research.143499.1) Latest published: 19 May 2025, 13:211 (https://doi.org/10.12688/f1000research.143499.3)

Revised Amendments from Version 1

Introduction: added more about cause, muscle function, more about muscle function scale and more about treatment.
Case I: added how ROM was measured and classification degree according to Kaplan et al.
Added to figure 3: Infants in general is righting the trunk to some extent in addition to righting the head, especially with higher scores.
Both cases clarifying about home training program
Case II added about taping: The taping was done by the physiotherapist once a week or more often if tape came off earlier. Taping is not easy for parents without experience, infants have a short neck and a lot of skin, so taping is challenging for the inexperienced.

Added in Discussion: more about muscle function and differential diagnosis. When torticollis does not concede with right or left CMT, it must be ensured that there is no other cause. There are several differential diagnoses, some of them more serious.
Added about long-term effect of KT is uncertain and needs to be further investigated. Two studies comparing CMT treatment with and without KT came to opposite conclusions about the effect of KT.
Conclusion rewritten: As bilateral CMT is rare and might be confusing, it needs to be further evaluated. Critical examination and documentation when evaluating CMT is important to give the right treatment when bilateral and also to make sure that a differential diagnosis is not missed by mistake. The need for longer follow-up for infants with bilateral CMT should be considered, to learn if there are any long-term problems.

Added some references.

See the author's detailed response to the review by Lucie Pelland
See the author's detailed response to the review by Nidhi Sharma and Parveen Kumar
See the author's detailed response to the review by Leo van Vlimmeren
See the author's detailed response to the review by Kimberly B. Castle

Introduction

CMT is a common congenital musculoskeletal anomaly among infants. It results from shortening or excessive contraction of the SCM muscle. One to four weeks after birth a SMT may be visible, which consists of fibrous tissue and disappears within a few months (Cheng, Tang and Chen, 1999; Kurtycz, Logrono, Hoerl and Heatley, 2000; Tang et al., 2002; Wei, Schwartz, Weaver and Orvidas, 2001; Ohman and Beckung, 2008). The head is typically tilted towards the affected muscle, and the chin is rotated towards the other side. The true etiology of CMT have been discussed; one hypothesis is that the condition could be the sequel of an intrauterine or perinatal compartment syndrome (Davis, Wenger, Mubarak, 1993), several studies are pointing in that direction (Bielski et al., 2006; Hardgrib, Rahbek, Möller-Madse and Maimburg, 2017; Lee et al., 2011a, 2011b; Pazonyi, Kun and Czeizel, 1982; Xiong et al., 2019). CMT can be divided into three groups:

SMT group with a clinically palpable sternomastoid tumor
MT group with tightness of the SCM muscle but no tumor
PT group with all the clinical features of torticollis, but without the tightness or tumor of the SCM muscle (Cheng et al., 2001).

For the SMT group, intrauterine or perinatal compartment syndrome is very likely to be the cause. Mandibular asymmetry is seen during examination, Fenton et al. found decreased ramal height ipsilateral to the affected SCM muscle (Fenton et al. 2018). It is possible that all three groups have the same cause, which is even more obvious in SMT and MT.

Infants with CMT have asymmetry in muscle function in the lateral flexors of the neck. The affected side is stronger than the other side (Öhman and Beckung, 2005; Ohman Mardbrink, Stensby and Beckung, 2011). Assessment of side-flexor musle function in lateral head righting can be evaluated with the Muscle Function Scale (MFS) (Ohman and Beckung, 2008; Kaplan, Coulter and Sargent, 2018). The MFS is found to be a reliable tool to test infants for CMT (Ohman, Nilsson and Beckung, 2009; Seager; French and Meldrum, 2019). It gives information on asymmetry in head righting response. If the infant tilts the head without limited ROM and no difference in scores on the MFS, other causes than CMT must be considered. There are several differential diagnoses of CMT and some of them can be potentially life threating (Zvi and Thompson, 2022). If an infant has an SMT on one side and tilts to the other side with higher MFS scores on that side, bilateral torticollis must be considered.

ROM in the neck can be measured with an arthrodial protractor (Cheng et al., 2001; Ohman and Beckung, 2008; Kaplan, Coulter and Sargent, 2018).

Treatment for CMT includes manual stretching, strengthening cervical muscles through positioning, handling and exercises isolating the weaker muscle, incorporating righting reactions in different positions. Developmental exercises should be incorporated to promote symmetrical movement in weight-bearing in different positions, as well as environmental adaptions and parent/caregiver education (Kaplan, Coulter and Sargent, 2018). Kinesiology taping (KT) can be used as a complementary treatment, to give an immediate relaxing effect (Ohman, 2012; Ohman, 2015).

Bilateral CMT is rare and not often reported, Matuszewski, Pietrzyk, Kandzierski and Wilczynsk (2017) reported a case of a child with bilateral torticollis, referred to the orthopedic department at the age of 12 years. The first symptoms appeared at preschool age. This child had severe limitations in range of motion and required bilateral surgery (Matuszewski, Pietrzyk, Kandzierski and Wilczynsk, 2017). Babu et al., presented a case report of a 19-year-old girl with congenital bilateral sternocleidomastoid contracture (Babu, Lee, Mahadev and Lee, 2009).

A few articles describing case reports of bilateral torticollis have not been published in English (Chiari, 1952; Kustos and Magdics, 1993; Shi et al., 2016; Shinoda and Yamada, 1969).

Case I

Case I is a Caucasian boy who came for a second opinion since the first examiner found it confusing, when the motion, muscle function/strength, and tilting of the head did not clearly fit left- or right-sided torticollis. On clinical examination in October 2020 when he was three and a half months of age, he had bilateral torticollis. On the right side, the patient had a SMT and some limitation in rotation towards the right side and in lateral flexion towards the left side, that is, the muscle on the right side was shortened. Case I had classification grade 3 according to Kaplan et al. CMT classification grades and decision tree for 0-12 months (Kaplan, Coulter and Sargent, 2018). When lying in the supine or prone position, he tilted the head to the right side and rotated to the left (Figure 1), which fit well with right-sided torticollis, SMT. While sitting with support, he tilted the head to the left (Figure 2) and was stronger in the lateral flexors on the left side (Figures 3 and 4), which fit well with a postural left-sided torticollis (PT). ROM in the neck rotation was measured with an arthrodial protractor and muscle function was evaluated using the MFS scale (Figure 5) (Ohman and Beckung, 2008; Ohman, Nilsson and Beckung, 2009).

Figure 1. Case I, in the prone position, showed lateral flexion of the head to the right and rotation to the left.

This is in agreement with right-sided torticollis. I confirm that I have obtained permission to use this image from the parents of the patient included in this presentation.

Figure 2. Case I in supported sitting position, lateral flexion of the head to the left and rotation to the right.

This is agreeable with left-sided torticollis. I confirm that I have obtained permission to use this image from the parents of the patient included in this presentation.

Figure 3. Case I test with the Muscle Function Scale (MFS) left side scoring 3, i.e., left side is stronger than the right side.

This is agreeable with left-sided torticollis Infants in general is righting the trunk to some extent in addition to righting the head, especially with higher scores. I confirm that I have obtained permission to use this image from the parents of the patient included in this presentation.

Figure 4. Case I test with the Muscle Function Scale (MFS) right side scoring 2, i.e., right side is weaker than the left side.

This is agreeable with left-sided torticollis. I confirm that I have obtained permission to use this image from the parents of the patient included in this presentation.

Figure 5. Muscle Function Scale.

When the Muscle Function Scale (MFS) is used, the infant is held in a vertical position and then lowered to the horizontal position in front of a mirror. The head position is observed and both sides are tested. Scores are given according to the head position in relation to the horizontal line. The infant must be observed with the head held in the same position for five seconds to obtain the score at that level. This figure has been reproduced/adapted from Ohman et al. (2009). Reprinted by permission of Informa UK Limited, trading as Taylor & Francis Group, www.tandfonline.com.

Treatment involved stretching of the SCM muscle on the right side performed by the physiotherapist three times per week. Home program with exercises; strengthening cervical muscles through positioning, handling and exrecises isolating the weaker SCM muscle. Incorporating rightning reactions in different positions. The treatment at that time gave quick results, equal strength in the lateral flexors of the neck, motion in lateral flexion, and only a marginal difference in rotation.

At the follow-up at two years of age, he had relatively good ROM and no head tilt, but he had an indication of a discreet muscular string on the right side. At the next follow-up at two and a half years of age, he had a discreet tendency to tilt the head to the right and was slightly stronger on the right side. The muscular string is still rather discreet and felt only when stretching the muscle. He has started treatment again, and the muscular string may worsen as he grows in height. The SCM muscle grows from about 4 cm in infants to 14 cm at 13 years of age, according to measures made by Jones (1968). Case I must be followed-up for a longer time, as there is a risk that he will need surgery later in life.

Case II

Case II is a Caucasian girl who was prenatally in breech presentation, and was turned by the healthcare provider some weeks before delivery. At the time of delivery, she was in a cephalic presentation and was fixed in the birth canal. In August 2020 at two and a half months of age, she came for clinical examination, referred for moderate brachycephaly. It was also obvious that the neck was affected, the SCM muscle was thickened bilaterally, and both active and passive rotations were affected. The head was held in flexion (Figure 6), and active rotation was severely limited to both sides, less than 45° bilaterally (Figure 7). Passive rotation was close to 90°, but with a clear stop, the mean normal range of rotation for infants is 110° (Ohman and Beckung, 2008). KT was used as a complement with a relaxing technique on both sides (Figure 8), and tape was applied across the sternocleidomastoid muscle on both sides. The taping was done by the physiotherapist once a week or more often if tape came off earlier. Taping is not easy for parents without experience, infants have a short neck and a lot of skin, so taping is challenging for the inexperienced. The parents worked with the home program several times every day with head control and exercises incorporated to promote symmetrical movement in different positions. When taped, it was easier for her to move the head with a greater range of motion. When she was about five months old, the problem was solved, and she had a good head position and active rotation of approximately 80° bilaterally and passive rotation >90° bilaterally. However, after reading the case reported by Matuszewski et al. (2017) I decided to check her again.

Figure 6. Front view of Case II, head in flexion due to bilateral torticollis.

I confirm that I have obtained permission to use this image from the parents of the patient included in this presentation.

Figure 7. Side view of Case II, head in flexion and only attempts to rotate the head.

I confirm that I have obtained permission to use this image from the parents of the patient included in this presentation.

Figure 8. Case II rotation of the head after application of kinesiology tape, relaxing technique cross over the sternocleidomastoid muscle.

I confirm that I have obtained permission to use this image from the parents of the patient included in this presentation.

At the age of nearly three years, she had an active rotation of 70° bilaterally and passive rotation of 90° bilaterally. Passive lateral flexion was full (i.e., ear to shoulder bilateral). Muscle function were assessed bilaterally according to the MFS scale she scored 4 bilaterally. The marginal flexion of the head might be within the normal span. I have decided to follow her once a year for at least some years to ensure that it stays good.

Discussion

Case I

The combination of SMT and PT on different sides has to my knowledge, not been reported. Even though it is rare, more cases probably exist as they are easy to miss if very mild. The combination of SMT and PT on different sides has to my knowledge, not been reported. Even though it is rare, more cases probably exist as they are easy to miss if very mild. It is important with rigorous examination and documentation to evaluate infants with torticollis, if it is bilateral CMT, the treatment must be adjusted. The MFS scale can be used in evaluating asymmetry in side-flexor muscle function in lateral head righting in PT. Healthy infants without CMT are not found to have any asymmetry on the MFS-scale (Ohman and Beckung, 2008). For example, in clinic infants with oculat roticollis show no difference on the MFS scale (author´s experience). Still photography is a reliable method for measuring habitual head deviation from midline in infants with CMT (Rahlin and Sarmiento, 2010). The examiner must follow progress attentively and carefully, and when needed adjust the treatment. When torticollis does not concide with right or left CMT, it must be ensured that there is no other cause. There are several differential diagnoses, some of them more serious (Zvi and Thompson, 2022).

As the muscle grows by about 10 cm during the child’s skeletal growth, in the first 13 years, it may be important to follow up during childhood. This will ensure the early discovery of any skeletal growth problems. In my experience, adults with neglected CMT have difficulty finding physicians or physiotherapists who understand that they have experienced CMT. Even with a clear muscular string and a typical head position, some adults do not receive a correct diagnosis and treatment. For a clinician with experience in children who need surgery for CMT, it is easy to recognize an adult with the same problem. However, bilateral torticollis is more confusing, and more knowledge of its long-term effects is needed.

KT was used as complementary treatment for case II and worked well for her. With relaxing technique, KT has an immediate relaxing effect on the tense muscle (Ohman, 2015). However, the long-term effect of KT is uncertain and needs to be further investigated (Kaplan, Coulter and Sargent, 2018). Two studies comparing CMT treatment with and without KT came to opposite conclusions about the effect of KT (Giray et al., 2017; Hussein, Ali and El-Meniawy, 2019).

Bilateral sternocleidomastoid tumors have been reported, with or without torticollis (Dangi and Gwasikoti, 2020; Kumar, Prabhu, Chattopadhayay and Nagendhar, 2003; Tufano, Tom and Austin, 1999). However, not much information is available on long-term results. Neck problems are not uncommon among adults, and we should be aware that there could have been an earlier bilateral torticollis that contributed to this problem.

Conclusions

As bilateral CMT is rare and might be confusing, it needs to be further evaluated. Critical examination and documentation when evaluating CMT is important to give the right treatment when bilateral and also to make sure that a differential diagnosis is not missed by mistake. The need for longer follow-up for infants with bilateral CMT should be considered, to learn if there are any long-term problems.

Consent

Written informed consent for publication of their clinical details and clinical images was obtained from the parents of the patients.

Data availability

All data underlying the results are available as part of the article and no additional source data are required.

Acknowledgements

Many thanks to the parents for allowing me to use their infants’ information and photos.

References

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Comments on this article Comments (1)

Version 3

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Version 2

VERSION 2 PUBLISHED 17 Jun 2024

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Discussion is closed on this version, please comment on the latest version above.

Author Response 19 May 2025

Anna Öhman, Health and Rehabilitation/Physiotherapy, University of Gothenburg, Gothenburg, Sweden

19 May 2025

Author Response

The focus is mainly to describe how bilateral torticollis can manifest.
The parents did not perform passive stretching, they stimulated active rotation.
In clinic for at least the last 10-15 ... Continue reading The focus is mainly to describe how bilateral torticollis can manifest.
The parents did not perform passive stretching, they stimulated active rotation.
In clinic for at least the last 10-15 years I always perform the stretching instead of the parents and the treatment time to get acceptable ROM is about one month. In several bigger studies by Cheng et al the stretching treatment is also done by experienced physiotherapist and not by parents.
My study comparing stretching done by physiotherapist and parents showed that if the stretching is done three time each week the result is much better than when parents stretch each day several times a day (Öhman A, Nilsson S, Beckung E. Stretching treatment for infants with Congenital Muscular Torticollis physiotherapist or parents, the treatment dilemma. PM&R 2010;2(12):1073-1079). Also, earlier studies by Cheng et al. have described stretching performed by physiotherapist three times per week and not by the parents (Cheng JC, Wong MW, Tang SP, Chen TM, Shum SL, Wong EM. Clinical determinants of the outcome of manual stretching in the treatment of congenital muscular torticollis in infants. A prospective study of eight hundred and twenty-one cases J Bone Joint Surg Am. 2001 83(5):679-87.; Cheng JCY, Tang SP, Chen TMK, Wong MWN, Wong EMC The clinical presentation and outcome of treatment of congenital muscular torticollis in infants--a study of 1,086 cases Pediatr Surg 2000 Jul;35(7):1091-6).. Why we expect parents to perform the stretching treatment is another discussion that should be taken, it is likely that the parents find this more stressful than the physiotherapist observe. (Oledzka MM, Sweeney JK, Evans-Rogers DL, Coulter C, Kaplan SL.Experiences of Parents of Infants Diagnosed With Mild or Severe Grades of Congenital Muscular Torticollis Pediatr Phys Ther. 2021 Oct 1;33(4):245.) For adults that need stretching treatment in i.e. a shoulder, nobody says that “this can you partner at home do”.
Case II recovered quickly but I do not find it unexpected quick, the muscle was thick but no SMT. There was no pain.
About figure 3 and MFS scale, with higher scores the infant usually also lifts up the trunk, MFS-scores cannot be compared with measuring active lateral flexion, if the active lateral flexion was the goal with MFS scale, I agree that the body should be in line. In theory you can want the body to be completely horizontal but that is not the reality in practice. When interpreting the MFS scale it is most important to read the text to each score, it is head in relation to a horizontal line that is scored and not in relation to the body’s position. We have to be pragmatic about how it works in practice.
I have asked to get figure 3 and figure 4 side by side, but administrator says it is not possible, I do not understand why as I changed size to make them fit side by side.

Secondary abnormalities are asked for, I am aware of these, but the aim of this article is not to cover everything that can occur. The two cases described had no extreme abnormalities.

Infants that have a sternomastoid tumor are of greater risk for need of surgery later in life. As the muscle grows from 4 cm as infant to 14 cm at 13 years of age (Jones P. Torticollis in infancy and childhood. Springfield, Illinois: Charles C Thomas; 1968) some children get limited motion at an older age. What chose of surgery cannot be predicted in advance and that is also a chose by the surgeon, rarely by the physiotherapist.
The focus is mainly to describe how bilateral torticollis can manifest.
The parents did not perform passive stretching, they stimulated active rotation.
In clinic for at least the last 10-15 years I always perform the stretching instead of the parents and the treatment time to get acceptable ROM is about one month. In several bigger studies by Cheng et al the stretching treatment is also done by experienced physiotherapist and not by parents.
My study comparing stretching done by physiotherapist and parents showed that if the stretching is done three time each week the result is much better than when parents stretch each day several times a day (Öhman A, Nilsson S, Beckung E. Stretching treatment for infants with Congenital Muscular Torticollis physiotherapist or parents, the treatment dilemma. PM&R 2010;2(12):1073-1079). Also, earlier studies by Cheng et al. have described stretching performed by physiotherapist three times per week and not by the parents (Cheng JC, Wong MW, Tang SP, Chen TM, Shum SL, Wong EM. Clinical determinants of the outcome of manual stretching in the treatment of congenital muscular torticollis in infants. A prospective study of eight hundred and twenty-one cases J Bone Joint Surg Am. 2001 83(5):679-87.; Cheng JCY, Tang SP, Chen TMK, Wong MWN, Wong EMC The clinical presentation and outcome of treatment of congenital muscular torticollis in infants--a study of 1,086 cases Pediatr Surg 2000 Jul;35(7):1091-6).. Why we expect parents to perform the stretching treatment is another discussion that should be taken, it is likely that the parents find this more stressful than the physiotherapist observe. (Oledzka MM, Sweeney JK, Evans-Rogers DL, Coulter C, Kaplan SL.Experiences of Parents of Infants Diagnosed With Mild or Severe Grades of Congenital Muscular Torticollis Pediatr Phys Ther. 2021 Oct 1;33(4):245.) For adults that need stretching treatment in i.e. a shoulder, nobody says that “this can you partner at home do”.
Case II recovered quickly but I do not find it unexpected quick, the muscle was thick but no SMT. There was no pain.
About figure 3 and MFS scale, with higher scores the infant usually also lifts up the trunk, MFS-scores cannot be compared with measuring active lateral flexion, if the active lateral flexion was the goal with MFS scale, I agree that the body should be in line. In theory you can want the body to be completely horizontal but that is not the reality in practice. When interpreting the MFS scale it is most important to read the text to each score, it is head in relation to a horizontal line that is scored and not in relation to the body’s position. We have to be pragmatic about how it works in practice.
I have asked to get figure 3 and figure 4 side by side, but administrator says it is not possible, I do not understand why as I changed size to make them fit side by side.

Secondary abnormalities are asked for, I am aware of these, but the aim of this article is not to cover everything that can occur. The two cases described had no extreme abnormalities.

Infants that have a sternomastoid tumor are of greater risk for need of surgery later in life. As the muscle grows from 4 cm as infant to 14 cm at 13 years of age (Jones P. Torticollis in infancy and childhood. Springfield, Illinois: Charles C Thomas; 1968) some children get limited motion at an older age. What chose of surgery cannot be predicted in advance and that is also a chose by the surgeon, rarely by the physiotherapist.
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Author details Author details

Health and Rehabilitation/Physiotherapy, University of Gothenburg, Gothenburg, Sweden

Anna Öhman
Roles: Conceptualization, Investigation, Writing – Original Draft Preparation

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No competing interests were disclosed.

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The author(s) declared that no grants were involved in supporting this work.

Article Versions (3)

version 3

Revised

Published: 19 May 2025, 13:211

https://doi.org/10.12688/f1000research.143499.3

version 2

Revised

Published: 17 Jun 2024, 13:211

https://doi.org/10.12688/f1000research.143499.2

version 1

Published: 21 Mar 2024, 13:211

https://doi.org/10.12688/f1000research.143499.1

© 2024 Öhman A. This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

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Öhman A. Bilateral congenital muscular torticollis in infants, report of two cases [version 2; peer review: 3 approved with reservations, 1 not approved]. F1000Research 2024, 13:211 (https://doi.org/10.12688/f1000research.143499.2)

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ApprovedThe paper is scientifically sound in its current form and only minor, if any, improvements are suggested

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Version 2

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Reviewer Report 09 May 2025

Nidhi Sharma, Maharishi Markandeshwar (Deemed to be University), Ambala, India

Parveen Kumar, Pal Healthcare, Haryana, India

Approved with Reservations

https://doi.org/10.5256/f1000research.166994.r376878

Summary of the Article
The author presents two case reports of infants with bilateral congenital muscular torticollis (CMT), a rare condition that can complicate diagnosis and management compared to the more common unilateral form. The manuscript aims to highlight the clinical presentation, assessment using the Muscle Function Scale (MFS), management including physiotherapy and kinesiology taping (KT), and short-term follow-up outcomes. The article also discusses differential diagnosis and emphasizes the need for long-term monitoring.
The author has provided clear descriptions of the cases, useful clinical photographs, and a practical discussion of challenges faced during diagnosis and treatment. The subject is relevant and contributes important observations to the relatively sparse literature on bilateral CMT.

ROM measurements are incompletely reported; secondary musculoskeletal features are not described; home program details need clarification.

Major Points to Address

Provide precise ROM values for active and passive movements.
Clarify the ROM measurement technique.
Explicitly report on secondary musculoskeletal features (e.g., head shape, trunk asymmetry).
Detail the home exercise program, particularly the stretching frequency.
Acknowledge or correct inconsistencies in MFS photograph interpretation.

Is the background of the cases’ history and progression described in sufficient detail?

Yes
Are enough details provided of any physical examination and diagnostic tests, treatment given and outcomes?

Partly
Is sufficient discussion included of the importance of the findings and their relevance to future understanding of disease processes, diagnosis or treatment?

Yes
Is the conclusion balanced and justified on the basis of the findings?

Yes

Competing Interests: No competing interests were disclosed.

Reviewer Expertise: Neuro-coordination, physiotherapy, Neurological rehabilitation, Musculoskeletal rehabilitation, Outcome measures, Quantitative and qualitative research

We confirm that we have read this submission and believe that we have an appropriate level of expertise to confirm that it is of an acceptable scientific standard, however we have significant reservations, as outlined above.

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Author Response 16 Jun 2025

Anna Öhman, Health and Rehabilitation/Physiotherapy, University of Gothenburg, Gothenburg, Sweden

16 Jun 2025

Author Response

Changed and added
Passive ROM in neck rotation was measured with an arthrodial protractor with the infant lying supine on the examination table with the shoulders stabilized. Case I had ... Continue reading Changed and added
Passive ROM in neck rotation was measured with an arthrodial protractor with the infant lying supine on the examination table with the shoulders stabilized. Case I had 95° towards the left side and 80° towards the right side. Lateral flexion was measured with the infant lying in supine position on a large protractor with the shoulders stabilized, 60° towards the left side and 70° towards the right side.

Case I had secondary features with mild plagiocefali on the left side and mild to moderate brachycefali.

Treatment involved stretching of the SCM muscle on the right side performed by the physiotherapist three times per week. Studies report shorter treatment periods when an experienced physiotherapist performs the stretching three times a week instead of parents performing daily stretching (Cheng et al 1999; Cheng et al 2000; Cheng et al 2001; Ohman, Nilsson and Beckung 2010). Home programmes include daily exercises done 3-5 times a day; active ROM in rotation, strengthening cervical muscles through positioning, handling and exercises isolating the weaker SCM muscle, incorporating righting reactions in different positions. The treatment at that time gave quick results, equal strength in the lateral flexors of the neck, motion in lateral flexion, and only a marginal difference in rotation. Plagiocephaly and brachycephaly were successfully treated with positioning.

Figure 3 is old figure 3 A and old figure 4 B
Figure 3. A. Case I test with the muscle function scale (MFS) left side scoring 3, i.e., left side is stronger than the right side. B. Case I test with the Muscle Function Scale (MFS) right side scoring 2, i.e., right side is weaker than the left side.
This is agreeable with left-sided torticollis Infants in general is righting the trunk to some extent in addition to righting the head, especially with higher scores.
Changed and added
Passive ROM in neck rotation was measured with an arthrodial protractor with the infant lying supine on the examination table with the shoulders stabilized. Case I had 95° towards the left side and 80° towards the right side. Lateral flexion was measured with the infant lying in supine position on a large protractor with the shoulders stabilized, 60° towards the left side and 70° towards the right side.

Case I had secondary features with mild plagiocefali on the left side and mild to moderate brachycefali.

Treatment involved stretching of the SCM muscle on the right side performed by the physiotherapist three times per week. Studies report shorter treatment periods when an experienced physiotherapist performs the stretching three times a week instead of parents performing daily stretching (Cheng et al 1999; Cheng et al 2000; Cheng et al 2001; Ohman, Nilsson and Beckung 2010). Home programmes include daily exercises done 3-5 times a day; active ROM in rotation, strengthening cervical muscles through positioning, handling and exercises isolating the weaker SCM muscle, incorporating righting reactions in different positions. The treatment at that time gave quick results, equal strength in the lateral flexors of the neck, motion in lateral flexion, and only a marginal difference in rotation. Plagiocephaly and brachycephaly were successfully treated with positioning.

Figure 3 is old figure 3 A and old figure 4 B
Figure 3. A. Case I test with the muscle function scale (MFS) left side scoring 3, i.e., left side is stronger than the right side. B. Case I test with the Muscle Function Scale (MFS) right side scoring 2, i.e., right side is weaker than the left side.
This is agreeable with left-sided torticollis Infants in general is righting the trunk to some extent in addition to righting the head, especially with higher scores.
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Author Response 16 Jun 2025

Anna Öhman, Health and Rehabilitation/Physiotherapy, University of Gothenburg, Gothenburg, Sweden

16 Jun 2025

Author Response

Changed and added
Passive ROM in neck rotation was measured with an arthrodial protractor with the infant lying supine on the examination table with the shoulders stabilized. Case I had ... Continue reading Changed and added
Passive ROM in neck rotation was measured with an arthrodial protractor with the infant lying supine on the examination table with the shoulders stabilized. Case I had 95° towards the left side and 80° towards the right side. Lateral flexion was measured with the infant lying in supine position on a large protractor with the shoulders stabilized, 60° towards the left side and 70° towards the right side.

Case I had secondary features with mild plagiocefali on the left side and mild to moderate brachycefali.

Treatment involved stretching of the SCM muscle on the right side performed by the physiotherapist three times per week. Studies report shorter treatment periods when an experienced physiotherapist performs the stretching three times a week instead of parents performing daily stretching (Cheng et al 1999; Cheng et al 2000; Cheng et al 2001; Ohman, Nilsson and Beckung 2010). Home programmes include daily exercises done 3-5 times a day; active ROM in rotation, strengthening cervical muscles through positioning, handling and exercises isolating the weaker SCM muscle, incorporating righting reactions in different positions. The treatment at that time gave quick results, equal strength in the lateral flexors of the neck, motion in lateral flexion, and only a marginal difference in rotation. Plagiocephaly and brachycephaly were successfully treated with positioning.

Figure 3 is old figure 3 A and old figure 4 B
Figure 3. A. Case I test with the muscle function scale (MFS) left side scoring 3, i.e., left side is stronger than the right side. B. Case I test with the Muscle Function Scale (MFS) right side scoring 2, i.e., right side is weaker than the left side.
This is agreeable with left-sided torticollis Infants in general is righting the trunk to some extent in addition to righting the head, especially with higher scores.
Changed and added
Passive ROM in neck rotation was measured with an arthrodial protractor with the infant lying supine on the examination table with the shoulders stabilized. Case I had 95° towards the left side and 80° towards the right side. Lateral flexion was measured with the infant lying in supine position on a large protractor with the shoulders stabilized, 60° towards the left side and 70° towards the right side.

Case I had secondary features with mild plagiocefali on the left side and mild to moderate brachycefali.

Treatment involved stretching of the SCM muscle on the right side performed by the physiotherapist three times per week. Studies report shorter treatment periods when an experienced physiotherapist performs the stretching three times a week instead of parents performing daily stretching (Cheng et al 1999; Cheng et al 2000; Cheng et al 2001; Ohman, Nilsson and Beckung 2010). Home programmes include daily exercises done 3-5 times a day; active ROM in rotation, strengthening cervical muscles through positioning, handling and exercises isolating the weaker SCM muscle, incorporating righting reactions in different positions. The treatment at that time gave quick results, equal strength in the lateral flexors of the neck, motion in lateral flexion, and only a marginal difference in rotation. Plagiocephaly and brachycephaly were successfully treated with positioning.

Figure 3 is old figure 3 A and old figure 4 B
Figure 3. A. Case I test with the muscle function scale (MFS) left side scoring 3, i.e., left side is stronger than the right side. B. Case I test with the Muscle Function Scale (MFS) right side scoring 2, i.e., right side is weaker than the left side.
This is agreeable with left-sided torticollis Infants in general is righting the trunk to some extent in addition to righting the head, especially with higher scores.
Competing Interests: No competing interests were disclosed. Close
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Reviewer Report 23 Jul 2024

Leo van Vlimmeren, Radboud University Medical Center, Nijmegen, The Netherlands

Approved with Reservations

https://doi.org/10.5256/f1000research.166994.r301321

This manuscript describes 2 cases with a rare, bilateral congenital muscular torticollis.
The focus seems to be mainly on the primary abnormalities resulting from the affected SCM on both sides: the passive, angular, cervical ROM (less on the active ROM) and on the muscle strength of the cervical lateral flexion (not on the rotation). This may be too limited in a description of the clinical manifestations.

Case 1 is recognizable. Also regarding the quick recovery. In the treatment, stretching of the SCM is mentioned by the physiotherapist 3 times a week. In my opinion, it is customary to have this carried out by the parents several times a day. Is this also done or was it a conscious choice to only stretch 3 times a week (which may not be effective, I think). The ROM is measured with the Arthrodial Protractor, but is not further explained in the description. The descriptions therefore remain vague and lack some conviction.

Case 2 is atypical for CMT. Such a rapid recovery from CMT (from age 2.5 to 5 months) is unlikely and seems to indicate a different etiology. Has cervical spine imaging been done? Could there have been cervical pain, for example caused by birth? Was there a (double-sided) SMT or was the SCM completely thickened or was there strand (string) formation?

As reviewer 2 already mentioned, the starting position of the child in Figure 3 is not correct. The trunk is not kept horizontal. This is not the case with all photos in Figure 5. This should be adjusted as this often leads to ambiguity in the implementation and interpretation of the MFS.

I miss the description of all secondary abnormalities, as are common with CMT:
• Shape of the head, face, jaws.
• Position of the ears, orbits, teeth.
• Position of the head at rest (lying or vertical position).
• Head deviation sideways.
• Shoulderstand: one- or two-sided elevation. Shoulder retraction.
• Spinal alignment. Scoliosis?
• Motor development. Asymmetry of trunk, arm and leg movements.
All these abnormalities can occur at a very young age and partly determine the choices of diagnosis and treatment.

Case 1 discusses a possible indication for surgery. On what basis would a decision be made to undergo surgery? Only primary or also secondary abnormalities due to CMT? And what should the operation consist? Mono or bipolar release? And what follow-up treatment would be necessary?

Is the background of the cases’ history and progression described in sufficient detail?

Yes
Are enough details provided of any physical examination and diagnostic tests, treatment given and outcomes?

Yes
Is sufficient discussion included of the importance of the findings and their relevance to future understanding of disease processes, diagnosis or treatment?

Yes
Is the conclusion balanced and justified on the basis of the findings?

Yes

Competing Interests: No competing interests were disclosed.

Reviewer Expertise: Pediatric physical therapy especially torticollis, skull deformation.

I confirm that I have read this submission and believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard, however I have significant reservations, as outlined above.

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Author Response 24 Jun 2025

Anna Öhman, Health and Rehabilitation/Physiotherapy, University of Gothenburg, Gothenburg, Sweden

24 Jun 2025

Author Response

Changed and added
Passive ROM in neck rotation was measured with an arthrodial protractor with the infant lying supine on the examination table with the shoulders stabilized. Case I had ... Continue reading Changed and added
Passive ROM in neck rotation was measured with an arthrodial protractor with the infant lying supine on the examination table with the shoulders stabilized. Case I had 95° towards the left side and 80° towards the right side. Lateral flexion was measured with the infant lying in supine position on a large protractor with the shoulders stabilized, 60° towards the left side and 70° towards the right side.

Treatment involved stretching of the SCM muscle on the right side performed by the physiotherapist three times per week. Studies report shorter treatment periods when an experienced physiotherapist performs the stretching three times a week instead of parents performing daily stretching (Cheng et al 1999; Cheng et al 2000; Cheng et al 2001; Ohman, Nilsson and Beckung 2010). Home programmes include daily exercises done 3-5 times a day; active ROM in rotation, strengthening cervical muscles through positioning, handling and exercises isolating the weaker SCM muscle, incorporating righting reactions.

When she was about five months old, the problem was solved, and she had a good head position and active rotation of approximately 80° bilaterally and passive rotation >90° bilaterally. Her recovery was relatively rapid possibly due to intense training by the parents at home.

Infants in general is righting the trunk to some extent in addition to righting the head, especially with higher scores.

Case I
Plagiocephaly and brachycephaly were successfully treated with positioning.

Case II
referred for moderate brachycephaly

Ther were no other abnormalities

Case I was not in need for surgery, I find no reason to add anything about surgery that not was needed.
Changed and added
Passive ROM in neck rotation was measured with an arthrodial protractor with the infant lying supine on the examination table with the shoulders stabilized. Case I had 95° towards the left side and 80° towards the right side. Lateral flexion was measured with the infant lying in supine position on a large protractor with the shoulders stabilized, 60° towards the left side and 70° towards the right side.

Treatment involved stretching of the SCM muscle on the right side performed by the physiotherapist three times per week. Studies report shorter treatment periods when an experienced physiotherapist performs the stretching three times a week instead of parents performing daily stretching (Cheng et al 1999; Cheng et al 2000; Cheng et al 2001; Ohman, Nilsson and Beckung 2010). Home programmes include daily exercises done 3-5 times a day; active ROM in rotation, strengthening cervical muscles through positioning, handling and exercises isolating the weaker SCM muscle, incorporating righting reactions.

When she was about five months old, the problem was solved, and she had a good head position and active rotation of approximately 80° bilaterally and passive rotation >90° bilaterally. Her recovery was relatively rapid possibly due to intense training by the parents at home.

Infants in general is righting the trunk to some extent in addition to righting the head, especially with higher scores.

Case I
Plagiocephaly and brachycephaly were successfully treated with positioning.

Case II
referred for moderate brachycephaly

Ther were no other abnormalities

Case I was not in need for surgery, I find no reason to add anything about surgery that not was needed.
Competing Interests: No competing interests were disclosed. Close
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Author Response 24 Jun 2025

Anna Öhman, Health and Rehabilitation/Physiotherapy, University of Gothenburg, Gothenburg, Sweden

24 Jun 2025

Author Response

Changed and added
Passive ROM in neck rotation was measured with an arthrodial protractor with the infant lying supine on the examination table with the shoulders stabilized. Case I had ... Continue reading Changed and added
Passive ROM in neck rotation was measured with an arthrodial protractor with the infant lying supine on the examination table with the shoulders stabilized. Case I had 95° towards the left side and 80° towards the right side. Lateral flexion was measured with the infant lying in supine position on a large protractor with the shoulders stabilized, 60° towards the left side and 70° towards the right side.

Treatment involved stretching of the SCM muscle on the right side performed by the physiotherapist three times per week. Studies report shorter treatment periods when an experienced physiotherapist performs the stretching three times a week instead of parents performing daily stretching (Cheng et al 1999; Cheng et al 2000; Cheng et al 2001; Ohman, Nilsson and Beckung 2010). Home programmes include daily exercises done 3-5 times a day; active ROM in rotation, strengthening cervical muscles through positioning, handling and exercises isolating the weaker SCM muscle, incorporating righting reactions.

When she was about five months old, the problem was solved, and she had a good head position and active rotation of approximately 80° bilaterally and passive rotation >90° bilaterally. Her recovery was relatively rapid possibly due to intense training by the parents at home.

Infants in general is righting the trunk to some extent in addition to righting the head, especially with higher scores.

Case I
Plagiocephaly and brachycephaly were successfully treated with positioning.

Case II
referred for moderate brachycephaly

Ther were no other abnormalities

Case I was not in need for surgery, I find no reason to add anything about surgery that not was needed.
Changed and added
Passive ROM in neck rotation was measured with an arthrodial protractor with the infant lying supine on the examination table with the shoulders stabilized. Case I had 95° towards the left side and 80° towards the right side. Lateral flexion was measured with the infant lying in supine position on a large protractor with the shoulders stabilized, 60° towards the left side and 70° towards the right side.

Treatment involved stretching of the SCM muscle on the right side performed by the physiotherapist three times per week. Studies report shorter treatment periods when an experienced physiotherapist performs the stretching three times a week instead of parents performing daily stretching (Cheng et al 1999; Cheng et al 2000; Cheng et al 2001; Ohman, Nilsson and Beckung 2010). Home programmes include daily exercises done 3-5 times a day; active ROM in rotation, strengthening cervical muscles through positioning, handling and exercises isolating the weaker SCM muscle, incorporating righting reactions.

When she was about five months old, the problem was solved, and she had a good head position and active rotation of approximately 80° bilaterally and passive rotation >90° bilaterally. Her recovery was relatively rapid possibly due to intense training by the parents at home.

Infants in general is righting the trunk to some extent in addition to righting the head, especially with higher scores.

Case I
Plagiocephaly and brachycephaly were successfully treated with positioning.

Case II
referred for moderate brachycephaly

Ther were no other abnormalities

Case I was not in need for surgery, I find no reason to add anything about surgery that not was needed.
Competing Interests: No competing interests were disclosed. Close
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Version 1

VERSION 1

PUBLISHED 21 Mar 2024

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Reviewer Report 09 May 2024

Lucie Pelland, Idaho State University, Pocatello, Idaho, USA

Not Approved

https://doi.org/10.5256/f1000research.157168.r264882

The topic is relevant to practice as bilateral torticollis is a rare clinical presentation. As stated by the author, a bilateral presentation can lead to uncertainty in diagnosis.

The Introduction could be improved in terms of the logical flow of information provided. As an example, possible causes of CMT are presented, including SCM tumor and intra-uterine or perinatal compartment syndrome. I suggest that these possible causes be related to the three clinical CMT groups presented. This would tie in how compartment syndrome relates to this classification. The author should also consider introducing the MFS in the introduction and present a rationale on how the MFS, within a standardized clinical assessment, could provide an approach to avoid a missed diagnosis of bilateral CMT. The introduction should end with the statement of aim provided in the abstract - which could be modified as follows: Our purpose was to describe the clinical presentation of bilateral CMT to improve diagnosis.

In the cases, there is no need to include the clinic where the clinical evaluation was performed. This information is provided in the author's contact. The language used should be formalized for scientific writing (e.g., 'I met this boy in October 2020' should be stated as 'On clinical examination in October 2020').
It might be clearer to align the figures for the two cases with the MFS and to provide the score for each Figure. For Case I, I would place Figures 3 and 4 side by side to more clearly show that the left SCM is stronger than the right. As well, in Figure 1, it is not clear if the head position may have been influenced by the asymmetric positioning of the upper limbs in prone support.
In case II, the HEP for parents is not clear - as well, was kinesiotaping applied at home? This would be important information for clinicians treating infants with bilateral CMT. For Case II, the figures do clearly show the forward head flexion. For clarity, I would suggest, again, that all positions in the MFS should be shown. This would help to problem solve the clinical presentation (which is the aim of the paper).
The quality of the images in Figure 5 needs to be improved.
The discussion repeats information that has been presented in the cases and in the introduction. I suggest that the discussion should focus on providing the structured approach, including the MFS, that should be used to avoid 'missing the diagnosis' of bilateral CMT. Treatment could be summarized in alignment with the clinical findings, providing a translation of findings to practice to improve patient outcomes.

General Comments:
1. There is a need for improving grammar and sentence structure throughout.
2. In the abstract, avoid using abbreviations that are only used once.
3. Specificity of terminology needs to be verified (e.g., in the abstract, 'how bilateral torticollis can appear in infants' would be better stated as 'how bilateral torticollis may present clinically'.
4. Once a term is abbreviated (e.g., SCM for sternocleidomastoid) it should then be used consistently.

Overall, this is a clinically relevant paper which focuses on an area of pediatric practice which requires further evidence.

Is the background of the cases’ history and progression described in sufficient detail?

Partly
Are enough details provided of any physical examination and diagnostic tests, treatment given and outcomes?

No
Is sufficient discussion included of the importance of the findings and their relevance to future understanding of disease processes, diagnosis or treatment?

No
Is the conclusion balanced and justified on the basis of the findings?

No

Competing Interests: No competing interests were disclosed.

Reviewer Expertise: Biomechanics, neuroscience, pediatrics

I confirm that I have read this submission and believe that I have an appropriate level of expertise to state that I do not consider it to be of an acceptable scientific standard, for reasons outlined above.

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Author Response 28 Jun 2024

Anna Öhman, Health and Rehabilitation/Physiotherapy, University of Gothenburg, Gothenburg, Sweden

28 Jun 2024

Author Response

Thanks for all valuable comments
made changes and added information, hopefully made the manuscript better.
New grammar check was also done
Competing Interests: No competing interests were disclosed.
Thanks for all valuable comments
made changes and added information, hopefully made the manuscript better.
New grammar check was also done
Thanks for all valuable comments
made changes and added information, hopefully made the manuscript better.
New grammar check was also done
Competing Interests: No competing interests were disclosed. Close
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Author Response 28 Jun 2024

Anna Öhman, Health and Rehabilitation/Physiotherapy, University of Gothenburg, Gothenburg, Sweden

28 Jun 2024

Author Response

Thanks for all valuable comments
made changes and added information, hopefully made the manuscript better.
New grammar check was also done
Competing Interests: No competing interests were disclosed.
Thanks for all valuable comments
made changes and added information, hopefully made the manuscript better.
New grammar check was also done
Thanks for all valuable comments
made changes and added information, hopefully made the manuscript better.
New grammar check was also done
Competing Interests: No competing interests were disclosed. Close
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Reviewer Report 02 May 2024

Kimberly B. Castle, University of North Georgia, Dahlonega, Georgia, USA

Approved with Reservations

https://doi.org/10.5256/f1000research.157168.r264886

This is a topic of interest to many pediatric physiotherapists and pediatricians. There is benefit in disseminating the information from these cases.

Details of the second case are much more specific than those of the first case. It is difficult to understand the exact extent of the first infant's passive limitation even with the photographs as there are no measures or estimates of limitations beyond the Muscle Function Scale.

As Dr. Ohman is the creator of the MFS, the evaluations using the tool are deemed to be accurate. However, the photos appear to show a difference in the trunk alignment of the infant when held to each side. Is the reader to assume that the infant is righting the trunk to some extent in addition to righting the head to the left (first MFS photo)? If not, is there a photo in which the therapist's hands support the trunk similarly that would allow comparison? Perhaps a brief explanation of the difference would be helpful for interpretation.

In the second case, was the flexed neck posture true neck flexion or rather a forward head posture with cervical flexion & capital extension? It is difficult to assess from the lateral photo and would be interesting to know if SCM were the only muscles that were tight or if there was increased tension in posterior neck musculature as well.

Finally, the discussion including typical growth in the muscular and skeletal growth throughout childhood was helpful. It would also be helpful to include a synopsis of how passive and active components impacted these children's posture and active movement depending on posture, position and the effects of gravity.

Is the background of the cases’ history and progression described in sufficient detail?

Partly
Are enough details provided of any physical examination and diagnostic tests, treatment given and outcomes?

Partly
Is sufficient discussion included of the importance of the findings and their relevance to future understanding of disease processes, diagnosis or treatment?

Partly
Is the conclusion balanced and justified on the basis of the findings?

Yes

Competing Interests: No competing interests were disclosed.

Reviewer Expertise: Pediatric physiotherapy with specific emphasis on core trunk and neck control including torticollis

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Author Response 28 Jun 2024

Anna Öhman, Health and Rehabilitation/Physiotherapy, University of Gothenburg, Gothenburg, Sweden

28 Jun 2024

Author Response

Author response:
Thanks for all valuable comments, there are a lot more to discuss about CMT,
Your review add some new thoughts.
Competing Interests: No competing interests were disclosed.
Author response:
Thanks for all valuable comments, there are a lot more to discuss about CMT,
Your review add some new thoughts.
Author response:
Thanks for all valuable comments, there are a lot more to discuss about CMT,
Your review add some new thoughts.
Competing Interests: No competing interests were disclosed. Close
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Author Response 08 Aug 2025

Anna Öhman, Health and Rehabilitation/Physiotherapy, University of Gothenburg, Gothenburg, Sweden

08 Aug 2025

Author Response

Changed and added
Passive ROM in neck rotation was measured with an arthrodial protractor with the infant lying supine on the examination table with the shoulders stabilized. Case I had ... Continue reading Changed and added
Passive ROM in neck rotation was measured with an arthrodial protractor with the infant lying supine on the examination table with the shoulders stabilized. Case I had 95° towards the left side and 80° towards the right side. Lateral flexion was measured with the infant lying in supine position on a large protractor with the shoulders stabilized, 60° towards the left side and 70° towards the right side.

MFS
Infants in general is righting the trunk to some extent in addition to righting the head, especially with higher scores.

Case II Ther was no obvious tension in other muscles. (not added).

Dependent on how short the muscle is it will more or less limit the ability to rotate towards the affected side and make it hard to sustain a straight position of the head. Torticollis in the neck may cause secondary scolios in the back in older children and adults (Min et al 2016; Kim, Yum and Shim, 2019; Choi et al 2024).
Changed and added
Passive ROM in neck rotation was measured with an arthrodial protractor with the infant lying supine on the examination table with the shoulders stabilized. Case I had 95° towards the left side and 80° towards the right side. Lateral flexion was measured with the infant lying in supine position on a large protractor with the shoulders stabilized, 60° towards the left side and 70° towards the right side.

MFS
Infants in general is righting the trunk to some extent in addition to righting the head, especially with higher scores.

Case II Ther was no obvious tension in other muscles. (not added).

Dependent on how short the muscle is it will more or less limit the ability to rotate towards the affected side and make it hard to sustain a straight position of the head. Torticollis in the neck may cause secondary scolios in the back in older children and adults (Min et al 2016; Kim, Yum and Shim, 2019; Choi et al 2024).
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Author Response 28 Jun 2024

Anna Öhman, Health and Rehabilitation/Physiotherapy, University of Gothenburg, Gothenburg, Sweden

28 Jun 2024

Author Response

Author response:
Thanks for all valuable comments, there are a lot more to discuss about CMT,
Your review add some new thoughts.
Competing Interests: No competing interests were disclosed.
Author response:
Thanks for all valuable comments, there are a lot more to discuss about CMT,
Your review add some new thoughts.
Author response:
Thanks for all valuable comments, there are a lot more to discuss about CMT,
Your review add some new thoughts.
Competing Interests: No competing interests were disclosed. Close
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Author Response 08 Aug 2025

Anna Öhman, Health and Rehabilitation/Physiotherapy, University of Gothenburg, Gothenburg, Sweden

08 Aug 2025

Author Response

Changed and added
Passive ROM in neck rotation was measured with an arthrodial protractor with the infant lying supine on the examination table with the shoulders stabilized. Case I had ... Continue reading Changed and added
Passive ROM in neck rotation was measured with an arthrodial protractor with the infant lying supine on the examination table with the shoulders stabilized. Case I had 95° towards the left side and 80° towards the right side. Lateral flexion was measured with the infant lying in supine position on a large protractor with the shoulders stabilized, 60° towards the left side and 70° towards the right side.

MFS
Infants in general is righting the trunk to some extent in addition to righting the head, especially with higher scores.

Case II Ther was no obvious tension in other muscles. (not added).

Dependent on how short the muscle is it will more or less limit the ability to rotate towards the affected side and make it hard to sustain a straight position of the head. Torticollis in the neck may cause secondary scolios in the back in older children and adults (Min et al 2016; Kim, Yum and Shim, 2019; Choi et al 2024).
Changed and added
Passive ROM in neck rotation was measured with an arthrodial protractor with the infant lying supine on the examination table with the shoulders stabilized. Case I had 95° towards the left side and 80° towards the right side. Lateral flexion was measured with the infant lying in supine position on a large protractor with the shoulders stabilized, 60° towards the left side and 70° towards the right side.

MFS
Infants in general is righting the trunk to some extent in addition to righting the head, especially with higher scores.

Case II Ther was no obvious tension in other muscles. (not added).

Dependent on how short the muscle is it will more or less limit the ability to rotate towards the affected side and make it hard to sustain a straight position of the head. Torticollis in the neck may cause secondary scolios in the back in older children and adults (Min et al 2016; Kim, Yum and Shim, 2019; Choi et al 2024).
Competing Interests: No competing interests were disclosed. Close
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Version 3

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Version 2

VERSION 2 PUBLISHED 17 Jun 2024

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Discussion is closed on this version, please comment on the latest version above.

Author Response 19 May 2025

Anna Öhman, Health and Rehabilitation/Physiotherapy, University of Gothenburg, Gothenburg, Sweden

19 May 2025

Author Response

The focus is mainly to describe how bilateral torticollis can manifest.
The parents did not perform passive stretching, they stimulated active rotation.
In clinic for at least the last 10-15 ... Continue reading The focus is mainly to describe how bilateral torticollis can manifest.
The parents did not perform passive stretching, they stimulated active rotation.
In clinic for at least the last 10-15 years I always perform the stretching instead of the parents and the treatment time to get acceptable ROM is about one month. In several bigger studies by Cheng et al the stretching treatment is also done by experienced physiotherapist and not by parents.
My study comparing stretching done by physiotherapist and parents showed that if the stretching is done three time each week the result is much better than when parents stretch each day several times a day (Öhman A, Nilsson S, Beckung E. Stretching treatment for infants with Congenital Muscular Torticollis physiotherapist or parents, the treatment dilemma. PM&R 2010;2(12):1073-1079). Also, earlier studies by Cheng et al. have described stretching performed by physiotherapist three times per week and not by the parents (Cheng JC, Wong MW, Tang SP, Chen TM, Shum SL, Wong EM. Clinical determinants of the outcome of manual stretching in the treatment of congenital muscular torticollis in infants. A prospective study of eight hundred and twenty-one cases J Bone Joint Surg Am. 2001 83(5):679-87.; Cheng JCY, Tang SP, Chen TMK, Wong MWN, Wong EMC The clinical presentation and outcome of treatment of congenital muscular torticollis in infants--a study of 1,086 cases Pediatr Surg 2000 Jul;35(7):1091-6).. Why we expect parents to perform the stretching treatment is another discussion that should be taken, it is likely that the parents find this more stressful than the physiotherapist observe. (Oledzka MM, Sweeney JK, Evans-Rogers DL, Coulter C, Kaplan SL.Experiences of Parents of Infants Diagnosed With Mild or Severe Grades of Congenital Muscular Torticollis Pediatr Phys Ther. 2021 Oct 1;33(4):245.) For adults that need stretching treatment in i.e. a shoulder, nobody says that “this can you partner at home do”.
Case II recovered quickly but I do not find it unexpected quick, the muscle was thick but no SMT. There was no pain.
About figure 3 and MFS scale, with higher scores the infant usually also lifts up the trunk, MFS-scores cannot be compared with measuring active lateral flexion, if the active lateral flexion was the goal with MFS scale, I agree that the body should be in line. In theory you can want the body to be completely horizontal but that is not the reality in practice. When interpreting the MFS scale it is most important to read the text to each score, it is head in relation to a horizontal line that is scored and not in relation to the body’s position. We have to be pragmatic about how it works in practice.
I have asked to get figure 3 and figure 4 side by side, but administrator says it is not possible, I do not understand why as I changed size to make them fit side by side.

Secondary abnormalities are asked for, I am aware of these, but the aim of this article is not to cover everything that can occur. The two cases described had no extreme abnormalities.

Infants that have a sternomastoid tumor are of greater risk for need of surgery later in life. As the muscle grows from 4 cm as infant to 14 cm at 13 years of age (Jones P. Torticollis in infancy and childhood. Springfield, Illinois: Charles C Thomas; 1968) some children get limited motion at an older age. What chose of surgery cannot be predicted in advance and that is also a chose by the surgeon, rarely by the physiotherapist.
The focus is mainly to describe how bilateral torticollis can manifest.
The parents did not perform passive stretching, they stimulated active rotation.
In clinic for at least the last 10-15 years I always perform the stretching instead of the parents and the treatment time to get acceptable ROM is about one month. In several bigger studies by Cheng et al the stretching treatment is also done by experienced physiotherapist and not by parents.
My study comparing stretching done by physiotherapist and parents showed that if the stretching is done three time each week the result is much better than when parents stretch each day several times a day (Öhman A, Nilsson S, Beckung E. Stretching treatment for infants with Congenital Muscular Torticollis physiotherapist or parents, the treatment dilemma. PM&R 2010;2(12):1073-1079). Also, earlier studies by Cheng et al. have described stretching performed by physiotherapist three times per week and not by the parents (Cheng JC, Wong MW, Tang SP, Chen TM, Shum SL, Wong EM. Clinical determinants of the outcome of manual stretching in the treatment of congenital muscular torticollis in infants. A prospective study of eight hundred and twenty-one cases J Bone Joint Surg Am. 2001 83(5):679-87.; Cheng JCY, Tang SP, Chen TMK, Wong MWN, Wong EMC The clinical presentation and outcome of treatment of congenital muscular torticollis in infants--a study of 1,086 cases Pediatr Surg 2000 Jul;35(7):1091-6).. Why we expect parents to perform the stretching treatment is another discussion that should be taken, it is likely that the parents find this more stressful than the physiotherapist observe. (Oledzka MM, Sweeney JK, Evans-Rogers DL, Coulter C, Kaplan SL.Experiences of Parents of Infants Diagnosed With Mild or Severe Grades of Congenital Muscular Torticollis Pediatr Phys Ther. 2021 Oct 1;33(4):245.) For adults that need stretching treatment in i.e. a shoulder, nobody says that “this can you partner at home do”.
Case II recovered quickly but I do not find it unexpected quick, the muscle was thick but no SMT. There was no pain.
About figure 3 and MFS scale, with higher scores the infant usually also lifts up the trunk, MFS-scores cannot be compared with measuring active lateral flexion, if the active lateral flexion was the goal with MFS scale, I agree that the body should be in line. In theory you can want the body to be completely horizontal but that is not the reality in practice. When interpreting the MFS scale it is most important to read the text to each score, it is head in relation to a horizontal line that is scored and not in relation to the body’s position. We have to be pragmatic about how it works in practice.
I have asked to get figure 3 and figure 4 side by side, but administrator says it is not possible, I do not understand why as I changed size to make them fit side by side.

Secondary abnormalities are asked for, I am aware of these, but the aim of this article is not to cover everything that can occur. The two cases described had no extreme abnormalities.

Infants that have a sternomastoid tumor are of greater risk for need of surgery later in life. As the muscle grows from 4 cm as infant to 14 cm at 13 years of age (Jones P. Torticollis in infancy and childhood. Springfield, Illinois: Charles C Thomas; 1968) some children get limited motion at an older age. What chose of surgery cannot be predicted in advance and that is also a chose by the surgeon, rarely by the physiotherapist.
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Kimberly B. Castle, University of North Georgia, Dahlonega, USA
Lucie Pelland, Idaho State University, Pocatello, USA
Leo van Vlimmeren, Radboud University Medical Center, Nijmegen, The Netherlands
Nidhi Sharma, Maharishi Markandeshwar (Deemed to be University), Ambala, India

Parveen Kumar, Pal Healthcare, Haryana, India

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Nidhi Sharma, Maharishi Markandeshwar (Deemed to be University), Ambala, India

Parveen Kumar, Pal Healthcare, Haryana, India

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Approved

Dear Authors,
You have done a commendable work. All the revisions have been made and no further comments are needed for the correction or modification in the present manuscript.

Thanks

Competing Interests

No competing interests were disclosed.

Reviewer Expertise

Neuro-coordination, physiotherapy, Neurological rehabilitation, Musculoskeletal rehabilitation, Outcome measures, Quantitative and qualitative research

We confirm that we have read this submission and believe that we have an appropriate level of expertise to confirm that it is of an acceptable scientific standard.

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Nidhi Sharma, Maharishi Markandeshwar (Deemed to be University), Ambala, India

Parveen Kumar, Pal Healthcare, Haryana, India

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Approved With Reservations

Provide precise ROM values for active and passive movements.
Clarify the ROM measurement technique.
Explicitly report on secondary musculoskeletal features (e.g., head shape, trunk asymmetry).
Detail the home exercise program, particularly the stretching frequency.
Acknowledge or correct inconsistencies in MFS photograph interpretation.

Is the background of the cases’ history and progression described in sufficient detail?

Yes
Are enough details provided of any physical examination and diagnostic tests, treatment given and outcomes?

Partly
Is sufficient discussion included of the importance of the findings and their relevance to future understanding of disease processes, diagnosis or treatment?

Yes
Is the conclusion balanced and justified on the basis of the findings?

Yes

Competing Interests

No competing interests were disclosed.

Reviewer Expertise

Neuro-coordination, physiotherapy, Neurological rehabilitation, Musculoskeletal rehabilitation, Outcome measures, Quantitative and qualitative research

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Author Response

16 Jun 2025

Anna Öhman, Health and Rehabilitation/Physiotherapy, University of Gothenburg, Gothenburg, Sweden

Changed and added
Passive ROM in neck rotation was measured with an arthrodial protractor with the infant lying supine on the examination table with the shoulders stabilized. Case I had 95° towards the left side and 80° towards the right side. Lateral flexion was measured with the infant lying in supine position on a large protractor with the shoulders stabilized, 60° towards the left side and 70° towards the right side.

Case I had secondary features with mild plagiocefali on the left side and mild to moderate brachycefali.

Treatment involved stretching of the SCM muscle on the right side performed by the physiotherapist three times per week. Studies report shorter treatment periods when an experienced physiotherapist performs the stretching three times a week instead of parents performing daily stretching (Cheng et al 1999; Cheng et al 2000; Cheng et al 2001; Ohman, Nilsson and Beckung 2010). Home programmes include daily exercises done 3-5 times a day; active ROM in rotation, strengthening cervical muscles through positioning, handling and exercises isolating the weaker SCM muscle, incorporating righting reactions in different positions. The treatment at that time gave quick results, equal strength in the lateral flexors of the neck, motion in lateral flexion, and only a marginal difference in rotation. Plagiocephaly and brachycephaly were successfully treated with positioning.

Figure 3 is old figure 3 A and old figure 4 B
Figure 3. A. Case I test with the muscle function scale (MFS) left side scoring 3, i.e., left side is stronger than the right side. B. Case I test with the Muscle Function Scale (MFS) right side scoring 2, i.e., right side is weaker than the left side.
This is agreeable with left-sided torticollis Infants in general is righting the trunk to some extent in addition to righting the head, especially with higher scores.

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23 Jul 2024 | for Version 2

Leo van Vlimmeren, Radboud University Medical Center, Nijmegen, The Netherlands

22 Views Cite this report Responses(1)

Approved With Reservations

Is the background of the cases’ history and progression described in sufficient detail?

Yes
Are enough details provided of any physical examination and diagnostic tests, treatment given and outcomes?

Yes
Is sufficient discussion included of the importance of the findings and their relevance to future understanding of disease processes, diagnosis or treatment?

Yes
Is the conclusion balanced and justified on the basis of the findings?

Yes

Competing Interests

No competing interests were disclosed.

Reviewer Expertise

Pediatric physical therapy especially torticollis, skull deformation.

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Author Response

24 Jun 2025

Anna Öhman, Health and Rehabilitation/Physiotherapy, University of Gothenburg, Gothenburg, Sweden

Changed and added
Passive ROM in neck rotation was measured with an arthrodial protractor with the infant lying supine on the examination table with the shoulders stabilized. Case I had 95° towards the left side and 80° towards the right side. Lateral flexion was measured with the infant lying in supine position on a large protractor with the shoulders stabilized, 60° towards the left side and 70° towards the right side.

Treatment involved stretching of the SCM muscle on the right side performed by the physiotherapist three times per week. Studies report shorter treatment periods when an experienced physiotherapist performs the stretching three times a week instead of parents performing daily stretching (Cheng et al 1999; Cheng et al 2000; Cheng et al 2001; Ohman, Nilsson and Beckung 2010). Home programmes include daily exercises done 3-5 times a day; active ROM in rotation, strengthening cervical muscles through positioning, handling and exercises isolating the weaker SCM muscle, incorporating righting reactions.

When she was about five months old, the problem was solved, and she had a good head position and active rotation of approximately 80° bilaterally and passive rotation >90° bilaterally. Her recovery was relatively rapid possibly due to intense training by the parents at home.

Infants in general is righting the trunk to some extent in addition to righting the head, especially with higher scores.

Case I
Plagiocephaly and brachycephaly were successfully treated with positioning.

Case II
referred for moderate brachycephaly

Ther were no other abnormalities

Case I was not in need for surgery, I find no reason to add anything about surgery that not was needed.

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No competing interests were disclosed.

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09 May 2024 | for Version 1

Lucie Pelland, Idaho State University, Pocatello, Idaho, USA

22 Views Cite this report Responses(1)

Not Approved

Is the background of the cases’ history and progression described in sufficient detail?

Partly
Are enough details provided of any physical examination and diagnostic tests, treatment given and outcomes?

No
Is sufficient discussion included of the importance of the findings and their relevance to future understanding of disease processes, diagnosis or treatment?

No
Is the conclusion balanced and justified on the basis of the findings?

No

Competing Interests

No competing interests were disclosed.

Reviewer Expertise

Biomechanics, neuroscience, pediatrics

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02 May 2024 | for Version 1

Kimberly B. Castle, University of North Georgia, Dahlonega, Georgia, USA

17 Views Cite this report Responses(2)

Approved With Reservations

Is the background of the cases’ history and progression described in sufficient detail?

Partly
Are enough details provided of any physical examination and diagnostic tests, treatment given and outcomes?

Partly
Is sufficient discussion included of the importance of the findings and their relevance to future understanding of disease processes, diagnosis or treatment?

Partly
Is the conclusion balanced and justified on the basis of the findings?

Yes

Competing Interests

No competing interests were disclosed.

Reviewer Expertise

Pediatric physiotherapy with specific emphasis on core trunk and neck control including torticollis

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Author Response

08 Aug 2025

Anna Öhman, Health and Rehabilitation/Physiotherapy, University of Gothenburg, Gothenburg, Sweden

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Competing Interests

No competing interests were disclosed.

Alongside their report, reviewers assign a status to the article:

Approved - the paper is scientifically sound in its current form and only minor, if any, improvements are suggested

Approved with reservations - A number of small changes, sometimes more significant revisions are required to address specific details and improve the papers academic merit.

Not approved - fundamental flaws in the paper seriously undermine the findings and conclusions

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Bilateral congenital muscular torticollis in infants, report of two cases

Abstract

Background

Case

Conclusions

Keywords

Revised Amendments from Version 1

Introduction

Case I

Figure 1. Case I, in the prone position, showed lateral flexion of the head to the right and rotation to the left.

Figure 2. Case I in supported sitting position, lateral flexion of the head to the left and rotation to the right.

Figure 3. Case I test with the Muscle Function Scale (MFS) left side scoring 3, i.e., left side is stronger than the right side.

Figure 4. Case I test with the Muscle Function Scale (MFS) right side scoring 2, i.e., right side is weaker than the left side.

Figure 5. Muscle Function Scale.

Case II

Figure 6. Front view of Case II, head in flexion due to bilateral torticollis.

Figure 7. Side view of Case II, head in flexion and only attempts to rotate the head.

Figure 8. Case II rotation of the head after application of kinesiology tape, relaxing technique cross over the sternocleidomastoid muscle.

Discussion

Case I

Conclusions

Consent

Data availability

Acknowledgements

References

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