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Chahaki E, Javanshir M, Saeeidi H, Taghdiri M. Investigating the Prevalence of Positional Plagiocephaly With 3D Scan in Children Under One Year of Age in Mofid Hospital. Func Disabil J 2021; 4 (1) : 33
URL: http://fdj.iums.ac.ir/article-1-144-en.html
1- Department of Orthotics and Prosthetics, Faculty of Rehabilitation, Iran University of Medical Science, Tehran, Iran.
2- Department of Orthotics and Prosthetics, Faculty of Rehabilitation, Iran University of Medical Science, Tehran, Iran. , saeedi.h@iums.ac.ir
3- Department of Neurology, Mofid Children Hospital, Shahid Beheshti University of Medical Sciences, Tehran, Iran.
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Introduction
Plagiocephaly is the most common skull deformity that eventually leads to the deformity of the head and the asymmetry of the baby’s skull bone [1]. Clinical signs of this deformity include unilateral flattening of the occipital bone on one side; changes in the position of the ear, in more severe cases; the protrusion of the forehead on the same side; the protrusion of the skull on the opposite side, and asymmetry in the face [2, 3]. According to these changes, the skull becomes the same as a parallelogram from the upper view [4]. Numerous risk factors play role in the development of plagiocephaly deformity; these factors can be divided into two categories: modifiable, non-modifiable. Modifiable risk factors can be promptly controlled through training and follow-up and their effect can be neutralized. Among the most important risk factors are male gender, multiple pregnancies and first delivery, delivery using assistive devices (vacuum and forceps), small uterus, large fetus, torticollis, neck disorders (limited neck movement tends to be in a fixed position), prolonged stay in the prone position in sleep, allocating inadequate time to be in the supine position during playing [5, 6, 7, 8, 9, 10,  11].
Sometimes in cases of severe deformity or rapid progression of deformity, surgical interventions for correction are on the agenda [12, 13]. Fortunately, conservative treatment is sufficient in most cases. The first line of treatment includes the teaching of positional therapy to the family, next, orthotic treatment is recommended when positional therapy is not enough. Teaching positional therapy to the family includes teaching parents how to position the baby’s head properly during sleeping, waking, breastfeeding, and carrying. This consultation should also specify the duration of the use of restraints, such as car seats or strollers during the treatment period [14].
A lot of evidence shows the delayed mental and motor development of this group of infants [2, 7, 8, 15, 16, 17, 18]. There is no report on the incidence and prevalence of this deformity in Iran, while many babies seem to have this problem. In other words, owing to the lack of scientific information about this anomaly, the problem remained unaddressed in the macro-planning of prevention and treatment of the Iranian society. On the other hand, the evaluation method and criteria are very effective in diagnosing and determining the severity of the deformity. Some previous studies [19] have used clinical scales to diagnose head deformity, while the validity and accuracy of such instruments depend on the examiner’s experience and are controversial. However, there is a significant relationship between the 3D and CT scans as diagnosing deformity. The present study aimed to define the prevalence of head deformities using the 3D scan and analyze the head parameters.

Materials and Methods
A total number of 90 children and their parents were included in this study. Inclusion criteria for children were the age of under 12 months and the lack of synostosis deformity. Initially, the parents were informed about the aim and procedure of the study. Then, they were asked to complete the consent form. After completing the child information forms and conducting a short interview to determine the risk factors, the child was prepared for 3D photography. First, cloth or nylon was placed on the child’s head so that the child’s face and ears were not covered. Then, we laid the child on the prepared table in the prone position and started scanning the head. During the scan, the minimum changes should be made in the position of the child’s head; this will be achieved since the scan time is very short. After analyzing the scanner and the therapist’s approval, the scanning phase is over. After preparing the 3D file, the landmarks were identified and the desired variables were measured in the prepared software. During this stage, first with different measurements on the child’s head, the amount of Cranial Vault Asymmetry Index (CVAI) and other parameters were measured to determine the severity of the deformity, then, information about risk factors was taken from the family.

3D scan
The 3-dimensional scanning device of the child’s head consists of two parts: hardware and software.

Hardware part
This part consists of a 3D scanner made by Microsoft (Xbox scanner, model: One’s 2016). The accuracy of this scanner at a distance of 50 cm is equal to 1 mm. During the scan, the therapist rotates the scanner around the child’s head to record information. Besides, the scanners are connected to a computer via a cable. This system is specifically designed to perform head-to-head scans and poses no danger to a child’s eyes.

Software part 
Special software has been designed that matches the scans of different images prepared by the scanner and finally forms a 3-dimensional image of the child’s head. This software can match the images prepared by the scanner. The second part of the software is responsible for landmarking and measuring the desired variables. The analyzed variable in this study included CVAI as a parameter for categorizing the deformity of the head.

Results
The scans of 90 children were included in the study. The prevalence of head deformity was 35% (32 children). We analyzed these 32 infants about the risk factors of deformity and the relationship between risk factors and deformation severity. Also, the Mean±SD age of the children was 7.82±3.03 months with an age range of 1.5 to 13 months. Moreover, the Mean±SD CVAI score was 6.01±3.35 percent. In this study, the Mean±SD score of The Overall Symmetry Ratio (OSR), Oblique Cranial Length Ratio (OCLR), and Cranial Index (CI) was 29.5±65.89, 106.19±4.17, and 114.11±1.32, respectively (Table 1).

The normality of the data was examined using the Kolmogorov-Smirnov and Shapiro-Wilk statistical tests. The results approved the normal distribution of the variables of age, CVAI, OCLR, score, and satisfaction, however, the data of the other variables were not normally distributed. 
The results show that the CVAI variable is negatively and significantly correlated with the variables of the duration of tummy position (laying on the stomach during playing) at P<0.001. Also, there is a significant positive correlation between CVAI and severity at the level of P<0.001. According to the result, the awareness variable has a significant negative correlation with the CVAI and disease severity variables at the level of P<0.01. Moreover, in a fixed position, the position of the neck is positively and significantly correlated with the CVAI variable and the disease severity at the level of P<0.001.
Relationship between the variance of disease severity and the studied variables 
Based on the Kruskal-Wallis test results, the variance of disease severity affects CVAI, OSR, and satisfaction (P<0.001) as well as the position of the neck in a fixed position, sleep on stomach duration, and the awareness of parents (P<0.05). Concerning the variable of satisfaction, the severity of the mild, moderate, and somewhat severe disease has a statistically significant relationship with CVAI. So, CVAI is the best index for estimating the severity of positional plagiocephaly.

Evaluation of the frequencies of positional deformity and quartiles based on CVAI index 
To examine this index, information, such as mean, middle, upper limit, lower limit, variance, standard deviation, and other criteria were examined. The highest frequency, the lowest value, and the highest value for this index were 6, 1.1, and 16.3, respectively. The results of quarter frequency showed that the numbers 3.2, 6.6, 7.7, and 10 were repeated twice. Besides, the results show that the average CVAI data is equal to 6.02 (Figure 1).

Positional deformity regression analysis based on CVAI index 
The analysis of variance was significant at the level of P<0.001, indicating the effectiveness of regression analysis. Also, the results show that there is a statistically significant relationship for the regression coefficients of the two variables of gender and OCLR at the levels of P<0.05 and P<0.01, respectively.

Discussion 
Various studies have obtained similar results as the current study. For example, in a study entitled Clinical Classification of Positional Plagiocephaly, Argenta measured the incidence of plagiocephaly in children of different age groups using digital cameras; the highest incidence rate was reported as 19.7% in four months infants. Measuring the amount of OCLR and CI, we obtained the incidence of plagiocephaly to be 35.5% in children under one year of age. Also, consistent with the present study, the mentioned study reported a CI greater than 93% and an OCLR greater than 106 in children with plagiocephaly deformity [8]. In a study, Arnivalla considered CVAI as a reliable criterion for measuring plagiocephaly. It was stated that if CVAI is between 3% to 7%, plagiocephaly is mild and shows a higher rate of up to 12% of severe plagiocephaly [3]. In the present study, CVAI was used to measure plagiocephaly, and it was observed that the average CVAI is about 6.01% (SD=3.35), while the CVAI was up to 16.30%, which indicates the severity of the disease.
The present study is consistent with the work of Mawji et al. 2010, which noted the relative superiority of the 3D imaging method for determining head deformities [20]. The 3D imaging provides more data in less time and can be a good alternative to the anthropometric method with a caliper. Also, consistent with the present study, Schaff et al. considered the CVAI index in the diagnosis of positional deformity. Given their relatively large sample size, it seems possible to match the results of their research with the present study. Generally, the criterion of CVAI and the use of 3D imaging is a good solution for the diagnosis of head deformities.
According to the Spearman nonparametric test in the present study, there is no significant correlation between gender, age, head circumference, and CI cranial index and other variables studied. On the other hand, the CVAI variable is negatively and significantly correlated with the variables of the duration of tummy position. Furthermore, the CVAI variable has a significant positive correlation with disease severity. The results show that awareness has a significant negative correlation with CVAI and disease severity. 
The position of the neck in a fixed position has a negative and significant correlation with CVAI. In a 2017 study, Aarnivala et al. examined the accuracy of measurements to quantify cranial asymmetry in plagiocephaly. The results showed that age had an insignificant effect on CVAI. Generally, the results showed that CVAI is a good scale for measuring plagiocephaly deformity [2]. In the present study, age and gender were insignificantly correlated with all of the parameters of asymmetry, which is in line with the results of the mentioned study.
In 2017, Huloka et al. reported a relationship between the duration of tummy time and severity and between awareness and severity, which are also mentioned in the present study [21]. A 2013 study by Maoji et al. found a commonality between the two studies regarding the 2.7-fold probability of head deformity in children who slept in the fixed position. Also, the present study observed a slightly higher degree of positional deformity in boys. Thus, in the case of the head in a fixed position, the result of the study can be generalized to the present study with a relative approximation. One advantage of the Maoji et al. research is the large sample size they reviewed, which makes it more likely to describe the results [22].
In 2012, Yoo et al. used cranial molding therapy and also the CVAI index to measure the degree of plagiocephaly deformity; they found that this treatment reduced the percentage of CVAI and increased parental satisfaction in using it [4]. In the present study, it was observed that the two variables of CVAI and parental satisfaction were significantly and negatively correlated with each other, which confirmed the results of this study.
The results of this study show that based on the Kruskal-Wallis test, the variance of disease severity affects CVAI, the variables of the neck in a fixed position, the duration of tummy time, and the awareness of parents. Concerning the variable of satisfaction, it is observed that the severity of the disease has a strong relationship with the increase in satisfaction. Moreover, the severity of mild, moderate, and somewhat severe disease has a statistically significant relationship with CVAI. 

Conclusion
The incidence of the deformity was estimated at 35.5% in children referred to Mofid hospital. Besides, the severity of plagiocephaly was assessed based on CVAI and classified as mild to moderate in severity for infants with head deformity. Also, among the mentioned indicators, the CVAI index showed more practical value than the other indicators. Based on the analysis of research data, sleeping in the same position was positively and significantly related to the severity of plagiocephaly, so it is better to change the head position during sleep. Also, there was a signwificant relationship between the parents’ awareness of positional deformity and also lying on their stomachs and the severity of plagiocephaly, so we need public education for parents about all positions of a child during sleeping, milking, playing, etc. 

Ethical Considerations
Compliance with ethical guidelines

This study was approved by the Ethics Committee of the Iran University of Medical Sciences. 

Funding
The paper was extracted from the MSc. thesis from  the first author at the Department of Orthotics and Prosthetics, School of Rehabilitation Sciences, Iran University of Medical Sciences, Tehran.

Authors' contributions
Data gathering: Esmaeil Chahaki; Supervision: Mohammad Ali  Javanshir; Methodology: Hassan saeedi; Investigation, Writing – review & editing: All authors.

Conflict of interest
The authors declare that there is no conflict of interest.

Acknowledgments
The authors would like to thank all parents and children of Mofid Hospital for their contributions to conduct research in a hospital.


References
  1. Matushita H, Alonso N, Cardeal DD, de Andrade FG. Major clinical features of synostotic occipital plagiocephaly: Mechanisms of cranial deformations. Childs Nerv Syst. 2014; 30(7):1217-24. [DOI:10.1007/s00381-014-2414-7] [PMID]
  2. Aarnivala H, Vuollo V, Heikkinen T, Harila V, Holmström L, Pirttiniemi P, et al. Accuracy of measurements used to quantify cranial asymmetry in deformational plagiocephaly. J Craniomaxillofac Surg. 2017; 45(8):1349-56. [DOI:10.1016/j.jcms.2017.05.014] [PMID]
  3. Freudlsperger C, Steinmacher S, Saure D, Bodem JP, Kühle R, Hoffmann J, et al. Impact of severity and therapy onset on helmet therapy in positional plagiocephaly. J Craniomaxillofac Surg. 2016; 44(2):110-5. [DOI:10.1016/j.jcms.2015.11.016] [PMID]
  4. Yoo HS, Rah DK, Kim YO. Outcome analysis of cranial molding therapy in nonsynostotic plagiocephaly. Arch Plast Surg. 2012; 39(4):338-44. [DOI:10.5999/aps.2012.39.4.338] [PMID] [PMCID]
  5. Kane AA, Mitchell LE, Craven KP, Marsh JL. Observations on a recent increase in plagiocephaly without synostosis. Pediatrics.1996; 97(6 Pt 1):877-85. [PMID]
  6. Rekate HL. Occipital plagiocephaly: A critical review of the literature. J Neurosurg. 1998; 89(1):24-30. [DOI:10.3171/jns.1998.89.1.0024] [PMID]
  7. Mitchell EA, Scragg R, Stewart AW, Becroft DM, Taylor BJ, Ford RP, et al. Results from the first year of the New Zealand cot death study. N Z Med J. 1991;104(906):71-6. [PMID]
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  9. Ripley CE, Pomatto J, Beals SP, Joganic EF, Manwaring KH, Moss SD. Treatment of positional plagiocephaly with dynamic orthotic cranioplasty. J Craniofac Surg. 1994; 5(3):150-9. [DOI:10.1097/00001665-199407000-00003] [PMID]
  10. Clarren SK, Smith DW, Hanson JW. Helmet treatment for plagiocephaly and congenital muscular torticollis. J Pediatr. 1979; 94(1):43-6. [DOI:10.1016/S0022-3476(79)80347-9] [PMID]
  11. Higginbottom MC, Jones KL, James HE. Intrauterine constraint and craniosynostosis. Neurosurgery. 1980; 6(1):39-44. [DOI:10.1227/00006123-198001000-00005] [PMID]
  12. Rogers GF, Oh AK, Mulliken JB. The role of congenital muscular torticollis in the development of deformational plagiocephaly. Plast Reconstr Surg. 2009; 123(2):643-52. [DOI:10.1097/PRS.0b013e318196b9be] [PMID]
  13. Golden KA, Beals SP, Littlefield TR, Pomatto JK. Sternocleidomastoid imbalance versus congenital muscular torticollis: Their relationship to positional plagiocephaly. Cleft Palate Craniofac J. 1999; 36(3):256-61. [DOI:10.1597/1545-1569_1999_036_0256_sivcmt_2.3.co_2] [PMID]
  14. Marchac A, Arnaud E, Di Rocco F, Michienzi J, Renier D. Severe deformational plagiocephaly: Long-term results of surgical treatment. J Craniofac Surg. 2011; 22(1):24-9. [DOI:10.1097/SCS.0b013e3181f7dd4a] [PMID]
  15. St John D, Mulliken JB, Kaban LB, Padwa BL. Anthropometric analysis of mandibular asymmetry in infants with deformational posterior plagiocephaly. J Oral Maxillofac Surg. 2002; 60(8):873-7. [DOI:10.1053/joms.2002.33855] [PMID]
  16. van Vlimmeren LA, Takken T, van Adrichem LNA, van der Graaf Y, Helders PJM, Engelbert RHH. Plagiocephalometry: A non-invasive method to quantify asymmetry of the skull; A reliability study. Eur J Pediatr. 2006; 165(3):149-57. [DOI:10.1007/s00431-005-0011-1] [PMID]
  17. Balan P, Kushnerenko E, Sahlin P, Huotilainen M, Näätänen R, Hukki J. Auditory ERPs reveal brain dysfunction in infants with plagiocephaly. J Craniofac Surg. 2002; 13(4):520-5. [DOI:10.1097/00001665-200207000-00008] [PMID]
  18. Miller RI, Clarren SK. Long-term developmental outcomes in patients with deformational plagiocephaly. Pediatrics. 2000; 105(2):E26. [DOI:10.1542/peds.105.2.e26] [PMID]
  19. van Vlimmeren LA, van der Graaf Y, Boere-Boonekamp MM, L’Hoir MP, Helders PJM, Engelbert RHH. Risk factors for deformational plagiocephaly at birth and at 7 weeks of age: A prospective cohort study. Pediatrics. 2007; 119(2):e408-18. [DOI:10.1542/peds.2006-2012] [PMID]
  20. Mawji A, Vollman AR, Fung T, Hatfield J, McNeil DA, Sauvé R. Risk factors for positional plagiocephaly and appropriate time frames for prevention messaging. Paediatr Child Health. 2014; 19(8):423-7. [DOI:10.1093/pch/19.8.423] [PMID] [PMCID]
  21. Siegenthaler MH. Methods to diagnose, classify, and monitor infantile deformational plagiocephaly and brachycephaly: A narrative review. J Chiropr Med. 2015; 14(3):191-204. [DOI:10.1016/j.jcm.2015.05.003] [PMID] [PMCID]
  22. Mulliken JB, Vander Woude DL, Hansen M, LaBrie RA, Scott RM. Analysis of posterior plagiocephaly: Deformational versus synostotic. Plast Reconstr Surg. 1999; 103(2):371-80. [DOI:10.1097/00006534-199902000-00003] [PMID]
Type of Study: Research | Subject: Prosthetics and Orthotics
Received: 2021/02/17 | Accepted: 2021/06/29 | Published: 2021/09/1

References
1. Matushita H, Alonso N, Cardeal DD, de Andrade FG. Major clinical features of synostotic occipital plagiocephaly: mechanisms of cranial deformations. Child's Nerv. Syst. 2014;30:1217-24. [DOI:10.1007/s00381-014-2414-7] [PMID]
2. Aarnivala, Henri, Ville Vuollo, Tuomo Heikkinen, Virpi Harila, Lasse Holmström, Pertti Pirttiniemi, and A Marita %J Journal of Cranio-Maxillofacial Surgery Valkama. 2017. "Accuracy of measurements used to quantify cranial asymmetry in deformational plagiocephaly." 45 (8):1349-1356. [DOI:10.1016/j.jcms.2017.05.014] [PMID]
3. Freudlsperger, Christian, Sahra Steinmacher, Daniel Saure, Jens P Bodem, Reinald Kühle, Jürgen Hoffmann, and Michael %J Journal of Cranio-Maxillofacial Surgery Engel. 2016. "Impact of severity and therapy onset on helmet therapy in positional plagiocephaly." 44 (2):110-115. [DOI:10.1016/j.jcms.2015.11.016] [PMID]
4. Yoo, Han-Su, Dong Kyun Rah, and Yong Oock %J Archives of Plastic Surgery Kim. 2012. "Outcome analysis of cranial molding therapy in nonsynostotic plagiocephaly." 39 (4):33 [DOI:10.5999/aps.2012.39.4.338] [PMID] [PMCID]
5. Kane AA, Mitchell LE, Craven KP, Marsh JL. Observations on a recent increase in plagiocephaly without synostosis. Pediatrics 1996;97:877-85.
6. Rekate HL. Occipital plagiocephaly: a critical review of the literature. J. Neurosurg. 1998;89:24-30. [DOI:10.3171/jns.1998.89.1.0024] [PMID]
7. Mitchell EA, Scragg R, Stewart AW, Becroft DM, Taylor BJ, Ford RP, et al. Results from the first year of the New Zealand cot death study. N. Z. Med. J. 1991;104:71-6.
8. Argenta L. Clinical classification of positional plagiocephaly. J. Craniofac. Surg. 2004;15:368-72. [DOI:10.1097/00001665-200405000-00004] [PMID]
9. Ripley CE, Pomatto J, Beals SP, Joganic EF, Manwaring KH, Moss SD. Treatment of positional plagiocephaly with dynamic orthotic cranioplasty. J. Craniofac. Surg. 1994;5:150-9. [DOI:10.1097/00001665-199407000-00003] [PMID]
10. Clarren SK, Smith DW, Hanson JW. Helmet treatment for plagiocephaly and congenital muscular torticollis. J. Pediatr. 1979;94:43-6. [DOI:10.1016/S0022-3476(79)80347-9]
11. Higginbottom MC, Jones KL, James HE. Intrauterine constraint and craniosynostosis. Neurosurgery 1980;6:39-44. https://doi.org/10.1097/00006123-198001000-00005 [DOI:10.1227/00006123-198001000-00005] [PMID]
12. Rogers GF, Oh AK, Mulliken JB. The role of congenital muscular torticollis in the development of deformational plagiocephaly. Plast. Reconstr. Surg. 2009;123:643-52. [DOI:10.1097/PRS.0b013e318196b9be] [PMID]
13. Golden KA, Beals SP, Littlereld TR, Pomatto JK. Sternocleidomastoid imbalance versus congenital muscular torticollis: their relationship to positional plagiocephaly. Cleft palate-craniofacial J. 1999;36:256-61. https://doi.org/10.1597/1545-1569(1999)036<0256:SIVCMT>2.3.CO;2 https://doi.org/10.1597/1545-1569_1999_036_0256_sivcmt_2.3.co_2 [DOI:10.1597/1545-1569(1999)0362.3.CO;2] [PMID]
14. Marchac A, Arnaud E, Di Rocco F, Michienzi J, Renier D. Severe deformational plagiocephaly: long-term results of surgical treatment. J. Craniofac. Surg. 2011;22:24-9. [DOI:10.1097/SCS.0b013e3181f7dd4a] [PMID]
15. St. John D, Mulliken JB, Kaban LB, Padwa BL. Anthropometric analysis of mandibular asymmetry in infants with deformational posterior plagiocephaly. J. Oral Maxillofac. Surg. 2002;60:873-7. [DOI:10.1053/joms.2002.33855] [PMID]
16. Van Vlimmeren LA, Takken T, Van Adrichem LNA, Van Der Graaf Y, Helders PJM, Engelbert RHH. Plagiocephalometry: a non-invasive method to quantify asymmetry of the skull; a reliability study. Eur. J. Pediatr. 2006;165:149-57. [DOI:10.1007/s00431-005-0011-1] [PMID]
17. Balan P, Kushnerenko E, Sahlin P, Huotilainen M, Näätänen R, Hukki J. Auditory ERPs reveal brain dysfunction in infants with plagiocephaly. J. Craniofac. Surg. 2002;13:520-5. [DOI:10.1097/00001665-200207000-00008] [PMID]
18. Miller RI, Clarren SK. Long-term developmental outcomes in patients with deformational plagiocephaly. Pediatrics 2000;105:e26-e26. [DOI:10.1542/peds.105.2.e26] [PMID]
19. Van Vlimmeren LA, van der Graaf Y, Boere-Boonekamp MM, L'Hoir MP, Helders PJM, Engelbert RHH. Risk factors for deformational plagiocephaly at birth and at 7 weeks of age: a prospective cohort study. Pediatrics 2007;119:e408-18. [DOI:10.1542/peds.2006-2012] [PMID]
20. Mawji A, Vollman AR, Fung T, Hatfield J, McNeil DA, Sauvé R. Risk factors for positional plagiocephaly and appropriate time frames for prevention messaging. Paediatr. Child Health 2014;19:423-7. [DOI:10.1093/pch/19.8.423] [PMID] [PMCID]
21. Siegenthaler MH. Methods to diagnose, classify, and monitor infantile deformational plagiocephaly and brachycephaly: a narrative review. J. Chiropr. Med. 2015;14:191-204. [DOI:10.1016/j.jcm.2015.05.003] [PMID] [PMCID]
22. Mulliken JB, Vander DLW, Hansen M, LaBrie RA, Scott RM. Analysis of posterior plagiocephaly: deformational versus synostotic. Plast. Reconstr. Surg. 1999;103:371-80. [DOI:10.1097/00006534-199902000-00003] [PMID]

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