School scoliosis screening by Moiré topography – Overview for 33 years in Miyazaki Japan

School scoliosis screening by Moiré topography – Overview for 33 years in Miyazaki Japan

Journal of Orthopaedic Science xxx (2018) 1e5 Contents lists available at ScienceDirect Journal of Orthopaedic Science journal homepage: http://www...

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Journal of Orthopaedic Science xxx (2018) 1e5

Contents lists available at ScienceDirect

Journal of Orthopaedic Science journal homepage: http://www.elsevier.com/locate/jos

Original Article

 topography e Overview for 33 School scoliosis screening by Moire years in Miyazaki Japan Hiroshi Kuroki a, b, *, Takuya Nagai b, Etsuo Chosa b, Naoya Tajima c a

Department of Orthopaedic Surgery, National Hospital Organization Miyazaki Higashi Hospital, Miyazaki, Japan Department of Orthopaedic Surgery, University of Miyazaki Faculty of Medicine, Miyazaki, Japan c Department of Orthopaedic Surgery, Nozaki Higashi Hospital, Miyazaki, Japan b

a r t i c l e i n f o

a b s t r a c t

Article history: Received 23 September 2017 Received in revised form 2 March 2018 Accepted 13 March 2018 Available online xxx

 topography in Background: Since 1981, we have performed school scoliosis screening (SSS) using Moire Miyazaki, Japan and attained a certain result in detecting scoliosis. However, this screening system was  topographic equipment. The purpose of discontinued due to cessation of repair and production of Moire  topography on the basis this study was to make clear both the results and the problems of SSS by Moire of our past 33 years' experiences. Methods: The subjects were 689,293 students (5th grade boys in 200,329, 5th grade girls in 191,919, 8th  topography between grade boys in 151,351, and 8th grade girls in 145,694) who were screened by Moire  topography, the discovery 1981 and 2013. The number of students received SSS, the positive rate of Moire rate of scoliosis greater than 20 , the reference rate to the second screening, and the positive predictive  topography to detect scoliosis greater than 20 were investigated. value of Moire  Results: The number of students received SSS achieved a peak in 1992. The positive rate of Moire topography and the discovery rate of scoliosis were highest in 8th grade girls. The reference rates to the second screening were 49.8% in 5th grade students and 41.4% in 8th grade students. The positive predictive values were 2.1% in 5th grade students and 7.6% in 8th grade students.  topography seemed to be effective in detecting scoliosis although both the Conclusion: SSS by Moire positive predictive value and the reference rate to the second screening were low. © 2018 Published by Elsevier B.V. on behalf of The Japanese Orthopaedic Association.

1. Introduction Adolescent idiopathic scoliosis (AIS) is a complex three dimensional developmental deformity of the spinal column and associated rib cage characterized by a lateral deviation and axial rotation [1e3]. Untreated cases of AIS may progress, and severe cases are at increased risk for various morbidity problems and mortality [4]. Although several procedures for operative treatment of AIS have evolved, the most favorable treatment is still based on early detection to complete the treatment conservatively without surgical procedures. In this regard, school scoliosis screening (SSS) is a powerful tool that can be used to identify children who may have deformity as well as those who may be at high risk for the disease

* Corresponding author. Department of Orthopaedic Surgery, National Hospital Organization Miyazaki Higashi Hospital, 4374-1 Tayoshi Ooaza, Miyazaki 880-0911 Japan. Fax: þ81 985 56-2257. E-mail address: [email protected] (H. Kuroki).

[5]. Since the effectiveness of brace treatment has been established with the results of the bracing in adolescent idiopathic scoliosis trial (BrAIST) study [6], reduction of the number of patients that progress to surgery is expected if scoliosis patients are discovered by SSS at an early stage and able to start proper brace treatment precociously.  topogIn Miyazaki, Japan, we have performed SSS using Moire raphy since 1981 and clearly come to feel the effectiveness of it in detecting scoliosis patients. In fact, in about 70% of cases, the primary reason for consultation to our scoliosis outpatient clinic was through SSS. And the mean Cobb angle at the first visit was statistically smaller in the scoliosis patients with SSS than those without SSS. Further, advanced AIS patients who had over 40  curve had not increased last 10 years. However, future SSS by Moire topography was discontinued due to recent cessation of repair and  topographic equipment. Alternatively, production of Moire compulsory school musculoskeletal screening (SMS) that includes the evaluation of the spine with inspection by school doctors who are not exclusively orthopaedic surgeons was initiated in 2016.

https://doi.org/10.1016/j.jos.2018.03.005 0949-2658/© 2018 Published by Elsevier B.V. on behalf of The Japanese Orthopaedic Association.

 topography e Overview for 33 years in Miyazaki Japan, Please cite this article in press as: Kuroki H, et al., School scoliosis screening by Moire Journal of Orthopaedic Science (2018), https://doi.org/10.1016/j.jos.2018.03.005

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H. Kuroki et al. / Journal of Orthopaedic Science xxx (2018) 1e5

The purpose of this study was to make clear both the results and  topography on the basis of our past 33 the problems of SSS by Moire years' experiences. 2. Materials and methods Our SSS program consists of two examinations. The first examination is inspection by school doctors or nurses for students in all  topographic evaluation by orthopaedic grades in addition to Moire surgeons for the 5th grade and the 8th grade students. The positive cases of the first examination are given a second examination of radiographic evaluation by orthopaedic surgeons (Fig. 1). The first examination was entirely funded by public expense in principle, whereas, the second screening was paid from own expense. The  topography include Moire  fringes and judgement criteria of Moire  fringes, differences more body appearance. In evaluation by Moire than 1.5 fringes (7.5 mm) are positive. On the other hand, body appearance is evaluated subjectively on laterality of body balance, shoulder line, waist line, and scapular height (Fig. 2). The subjects enrolled in this study were 689,293 students who  topography between 1981 and 2013 in were screened by Moire Miyazaki Japan. They consisted of 5th grade boys in 200,329, 5th grade girls in 191,919, 8th grade boys in 151,351, and 8th grade girls  screening, the in 145,694. The number of students received Moire  topography, the discovery rate of scoliosis positive rate of Moire greater than 20 , the reference rate to the second screening, and the  topography to detect scoliosis positive predictive value of Moire greater than 20 were investigated. This study was approved by our institutional review board (approval number 29-14). And all procedures were in accordance with the Helsinki declaration. 3. Results In the early period after initiating SSS, the number of students  screening were less than 10,000 because they had received Moire to be charged a fee by themselves. After the public expense was allowed, the participants rapidly increased. In 1992, it reached over 29,000. Since then, however, it gradually decreased due to a declining birthrate (Fig. 3).

 topography were 6.11% in The average positive rates of Moire 8th grade girls, 5.22% in 5th grade girls, 2.89% in 5th grade boys, and 2.15% in 8th grade boys (Fig. 4). The average discovery rates of scoliosis were 0.24% in 8th grade girls, 0.07% in 5th grade girls, 0.03% in 8th grade boys, and 0.01% in 5th grade boys (Fig. 5). The average reference rates to the second screening were 50.37% in 5th grade girls, 48.64% in 5th grade boys, 44.39% in 8th grade boys, and 40.37% in 8th grade girls (Fig. 6). The average positive predictive values were 9.54% in 8th grade girls, 2.91% in 8th grade boys, 2.83% in 5th grade girls, and 0.89% in 5th grade boys (Table 1). 4. Discussion SSS has been established as a valuable method for early detection of the disease and prophylaxis for severe deformity [7,8]. The goal of SSS is to detect scoliosis at an early stage when deformity is likely to go unnoticed and when there is an opportunity for a less invasive method of treatment, or less surgery, than would otherwise be the case [9e11]. SSS has been practiced widely in the United States (US) and throughout the world for nearly 60 years [9]. To date, the efficacy of SSS has not been established sufficiently because of a lack of randomized, controlled trials. Some studies concluded that SSS is effective, whereas others questioned the effectiveness or even considered such screening to be unethical [12]. The United States Preventive Services Task Force (USPSTF) initially released an opinion on SSS in 1996 stating that there was insufficient evidence to make a recommendation for or against routine SSS [13]. However, in 2004, they changed their position and recommended against the routine screening of asymptomatic adolescents for idiopathic scoliosis [14]. In contrast, in 2008, the American Academy of Orthopaedic Surgeons (AAOS), the Scoliosis Research Society (SRS), the Pediatric Orthopaedic Society of North America (POSNA), and the American Academy of Pediatrics (AAP) issued an information statement indicating that they did not support any recommendations against SSS by USPSTF, given the available literature. Despite mounting evidence throughout the 1990s that SSS was ineffective, costly, and potentially harmful

Fig. 1. Screening system for scoliosis in Miyazaki Japan. The school screening system for scoliosis in Miyazaki Japan consisted of two examinations. The first examination was  topography for all of the 5th and 8th grades of school students. The inspection by school physicians or nurses for students in all grades of school students in addition to Moire positive cases of the first examination were given a second examination of radiographic evaluation by orthopaedists. Then, they were judged normal, need to be observation, and need to be treatment.

 topography e Overview for 33 years in Miyazaki Japan, Please cite this article in press as: Kuroki H, et al., School scoliosis screening by Moire Journal of Orthopaedic Science (2018), https://doi.org/10.1016/j.jos.2018.03.005

H. Kuroki et al. / Journal of Orthopaedic Science xxx (2018) 1e5

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 topography. a. A normal case has symmetric Moire  fringes and no laterality of body shape. b. Moire  fringes of an abnormal case show Fig. 2. School scoliosis screening by Moire  topographic equipment in position the subject is standing on the tilting frame with natural obvious laterality and waist line is also asymmetric. c. A back picture is taken with Moire posture.

 screening. The number of students received Moire  screening were less than 10,000 in the early period when a fee was charged at their Fig. 3. Number of students received Moire own expense. After the public expense was allowed, the participants rapidly increased and reached its peak about 29,000 in 1992. However, since then, it gradually decreased due to a declining birthrate.

 topography. The average positive rates of Moire  topography (PRMT) were 6.11% in 8th grade girls, 5.22% in 5th grade girls, 2.89% in 5th grade boys, and Fig. 4. Positive rate of Moire 2.15% in 8th grade boys. Although there was quite a bit variation in years, the PRMT was highest in 8th grade girls.

 topography e Overview for 33 years in Miyazaki Japan, Please cite this article in press as: Kuroki H, et al., School scoliosis screening by Moire Journal of Orthopaedic Science (2018), https://doi.org/10.1016/j.jos.2018.03.005

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H. Kuroki et al. / Journal of Orthopaedic Science xxx (2018) 1e5

Fig. 5. Discovery rate of scoliosis greater than 20 . The average discovery rate of scoliosis (DRS) greater than 20 were 0.24% in 8th grade girls, 0.07% in 5th grade girls, 0.03% in 8th grade boys, and 0.01% in 5th grade boys. Although there was quite a bit variation in years, the DRS was highest in 8th grade girls.

Fig. 6. Reference rate to the second screening. The average reference rates to the second screening (RRSS) were 50.37% in 5th grade girls, 48.64% in 5th grade boys, 44.39% in 8th grade boys, and 40.37% in 8th grade girls. Although there was quite a bit variation in years, the RRSS was higher in 5th grade students than in 8th grade students.

Table 1  topography to detect scoliosis greater than 20 . Positive predictive value of Moire Grade

5th grade boys

5th grade girls

8th grade boys

8th grade girls

PPV(%)

0.89

2.83

2.91

9.54

PPV: Positive predictive value.

psychologically, more and more US states instituted SSS [15]. By 2003, 21 states had legislated SSS, 11 states recommended SSS without legislation and the remaining states either had volunteer screening or recommended the avoidance of screening in schools [16]. In 2013, the SRS international task force began pursuing a consensus based on careful analysis of a recent critical review work of the literature on SSS, and performed using a conceptual framework of analysis focusing on five main dimensions: technical, clinical, program, cost, and treatment effectiveness. They concluded that there is moderate evidence that SSS allows for the detection and referral of patients at an earlier stage of the clinical course, and that there is low evidence suggesting that scoliosis patients detected by screening are less likely to need surgery than those

who did not undergo screening [17]. The USPSTF recently changed their position again that the current evidence is insufficient to assess the balance of benefits and harms of SSS in children and adolescents aged 10e18 years [18]. In Japan, SSS is mandated by law [16]. Revision of the law on school health in 1979 facilitated the implementation of SSS. The FBT and the measurement of ATR with a scoliometer were readily employed in some communities as the primary screening method  topography for students while other areas primarily used the Moire in specific grades. In the Hiroshima prefecture, the Silhouetter  topography, was first introsystem, which is similar to the Moire duced [19]. The students with positive judgement in the primary screening were referred to a second screening using radiography.  topography that There are some drawbacks in our SSS by Moire are originated from the screening system (cost and accuracy), the subjects (refusal), and the screening itself (psychological burden). With respect to the cost of SSS, the estimated cost of the primary  topography per person was 500 JPY (5.5 USD: screening by Moire an assumed exchange rate of 110 JPY to the 1 USD) [20]. The estimated cost of the secondary screening by radiography per person

 topography e Overview for 33 years in Miyazaki Japan, Please cite this article in press as: Kuroki H, et al., School scoliosis screening by Moire Journal of Orthopaedic Science (2018), https://doi.org/10.1016/j.jos.2018.03.005

H. Kuroki et al. / Journal of Orthopaedic Science xxx (2018) 1e5

was 10,000 JPY (110 USD) [20]. If 20,000 subjects have a primary screening a year, total cost is running at 10,000,000 JPY (110,000 USD). And if 1000 subjects have a secondary screening a year, total cost is also running at 10,000,000 JPY (110,000 USD). The cost issue is still highly controversial because the evaluative standard about cost-effectiveness has not been clearly established yet. The accuracy of screening methods includes problems. The  topography to predictive value of our primary screening by Moire detect over 20 curve was 2.1% in 5th grade students and 7.6% in the 8th grade students, respectively. In the previous work of  Kuroki et al., one reason for a false negative result of Moire topography was the difficulty evaluating a lumbar deformity  fringes at the lumbar level were crowded and because the Moire lack an apex of fringes [21]. Aside this point, the accuracy of screening fluctuates greatly depending on the screeners' decision  topography is strictly although the judgement criteria of Moire stipulated. Actually, screeners were replaced once every several years and positive rate of Moire topography was somewhat varied from year to year. Increase in the false-positive rate will be directly linked to rise in radiation exposure for normal subjects. On the other hand, increase in the false-negative rate will result in missing an opportunity to initiate the early treatment for scoliosis patients. Therefore, it is mandatory to improve the accuracy of the screening procedures. It is serious issue for subject to refuse the second screening despite judgement of abnormal findings in the primary screening due to lack of risk awareness. In our series, reference rate to the second screening was only 49.4% in 5th grade students and 41.4% in the 8th grade students, respectively. Further, Hines et al. [22] pointed a potential anxiety in school scoliosis screening. They stated through their cross-sequential study that children and their parents referred through SSS program experienced significantly elevated levels of state-anxiety, and this anxiety significantly decreased for the false-positive referrals, yet remained heightened for the true-positive referrals. It is essential to implement SSS without disbelief and anxiety by means of preliminary provision of accurate knowledge about scoliosis management. At present, SSS in Japan is currently at a turning point. Hereafter, reestablishment of SSS must be accomplished using SMS with the FBT alone for students who are fully dressed because the traditional school screening systems specific to scoliosis will finish in the near future. SRS insists that the scoliometer is currently the best tool available for SSS because the FBT results in an unacceptable number of false-negative results [17]. The use of a scoliometer can surely make the simplest, quickest, most reliable, and least expensive objective evaluation of trunk rotation possible. Indeed, screening with a scoliometer has the same or better accuracy than that with  topographic equipment [23]. Meanwhile, the burden on the Moire screeners is greater because direct examination is necessary. In the next couple of years, the results of SMS as well as cost-effectiveness will be analyzed in detail, and should be refined to reduce falsepositive and false-negative findings. In addition, the contents of the screening program should be reconsidered. The selection of the subject per se and the alteration of judgement criteria, depending on the sex or maturation of the subjects, may be preferable to increase the accuracy of the program and lighten the burden on the screeners. The SRS international task force stated that females should be screened twice, at ages 10 and 12, and boys should be screened once, at age 13 or 14. In addition, there is moderate evidence to recommend referral with scoliometer values greater than 5 [17]. Further, activities awareness promoting of scoliosis for the general public are indispensable. Dissemination of accurate

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knowledge about the importance of SSS can reduce the number of individual opposed to receiving SSS and consequently improve the scoliosis discovery rate at in an early stage. Distribution of brochures about scoliosis is also a valid and feasible option. In the future, an optimal SSS program should be established based on these factors to contribute the early detection and the suitable management of scoliosis. Conflict of interest The authors declare that they have no conflict of interest. References [1] Stokes IA. Three-dimensional terminology of spinal deformity. a report presented to the scoliosis research society by the scoliosis research society working group on 3-D terminology of spinal deformity. Spine 1994 Jan 15;19(2):236e48. [2] Adobor RD, Riise RB, Sørensen R, Kibsgård TJ, Steen H, Brox JI. Scoliosis detection, patient characteristics, referral patterns and treatment in the absence of a screening program in Norway. Scoliosis 2012 Oct 25;7(1):18. [3] Carlson BB, Burton DC, Asher MA. Comparison of trunk and spine deformity in adolescent idiopathic scoliosis. Scoliosis 2013 Jan 25;8(1):2. [4] Fong DYT, Lee CF, Cheung KMC, Cheng JCY, Ng BKW, Lam TP, Mak KH, Yip PSF, Luk KDK. A meta-analysis of the clinical effectiveness of school scoliosis screening. Spine 2010 May 1;35(10):1061e71. [5] Soucacos PN, Soucacos PK, Zacharis KC, Beris AE, Xenakis TA. School-screening for scoliosis: a prospective epidemiological study in northwestern and central Greece. J Bone Jt Surg Am 1997 Oct;79(10):1498e503. [6] Weinstein SL, Dolan LA, Wright JG, Dobbs MB. Effects of bracing in adolescents with idiopathic scoliosis. N Engl J Med 2013 Oct 17;369(16):1512e21. [7] Smyrnis PN, Valavanis J, Alexopoulos A, Siderakis G, Giannestras NJ. School screening for scoliosis in Athens. J Bone Jt Surg Br 1979 May;61(2):215e7. [8] Lonstein JE. Screening for spinal deformities in Minnesota schools. Clin Orthop Relat Res 1977 Jul-Aug;(126):33e42. [9] Bunnell WP. Selective screening for scoliosis. Clin Orthop Relat Res 2005 May;(434):40e5.  topography in scoliosis: its accuracy [10] Daruwalla JS, Balasubramaniam P. Moire in detecting the site and size of the curve. J Bone Jt Surg Br 1985 Mar;67(2): 211e3. [11] Grivas TB, Vasiliadis E, Savvidou OD, Triantafyllopoulos G. What a school screening program could contribute in clinical research of idiopathic scoliosis aetiology. Disabil Rehabil 2008;30(10):752e62. [12] Bunge EM, Juttmann RE, van Biezen FC, Creemers H, HazebroekKampschreur AAJM, Luttmer BCF, Wiegersma PA, de Koning HJ. Estimating the effectiveness of screening for scoliosis: a case-control study. Pediatrics 2008 Jan;121(9):9e14. [13] US Preventive Services Task Force (USPSTF). Screening for adolescent idiopathic scoliosis: policy statement. JAMA 1993 May 26;269(20):2664e6. [14] US Preventive Services Task Force (USPSTF). Screening for idiopathic scoliosis in adolescents: recommendation statement. Agency for Healthcare Research and Quality (AHRQ) Pub; November 2004. No. 05-0568-A. [15] Linker B. A dangerous curve: the role of history in American's scoliosis screening programs. Am J Publ Health 2012 Apr;102(4):606e16. [16] Grivas TB, Wade MH, Negrini S, O'Brien JP, Maruyama T, Hawes MC, Rigo M, Weiss HR, Kotwicki T, Vasiliadis ES, Sulam LN, Neuhous T. SOSORT consensus paper: school screening for scoliosis: where are we today? Scoliosis 2007 Nov 26;2(1):17. [17] Labelle H, Richards BS, Kleuver MD, Grivas TB, Luk KDK, Wong HK, Thometz J, jour M, Turgeon I, Fong DY. Screening for adolescent idiopathic scoliBeause osis: an information statement by the scoliosis research society international task force. Scoliosis 2013 Oct 31;8(1):17. [18] US Preventive Services Task Force (USPSTF). Screening for adolescent idiopathic scoliosis: US preventive services task force recommendation statement. JAMA 2018 Jan 9;319(2):165e72. [19] Izumi YA. Mobile Silhouetter system for mass screening. J Jpn Scoliosis Soc 2004;19(1):123e9 [in Japanese)]. [20] Kuroki H, Tajima N, Watanabe S. The current status and problems of school screening for scoliosis: from the results of screening program in Miyazaki prefecture. Orthop Surg Traumatol 2001;44(1):33e9 [in Japanese)].  [21] Kuroki H, Kubo S, Chosa E, Tajima N. Verification of school screening by Moire topography. J Jpn Scoliosis Soc 2008;23(1):14e20 [in Japanese)]. [22] Hines T, Roland S, Nguyen D, Kennard B, Richard H, Hughes CW, McClintock SM, Ramo B, Herring T. School scoliosis screenings: family experiences and potential anxiety after orthopaedic referrals. Spine 2015 Nov;40(21):E1135e43. [23] Chowanska J, Kotwicki T, Rosadzinski K, Sliwinski Z. School screening for scoliosis: can surface topography replace examination with scoliometer? Scoliosis 2012 Apr 3;7(1):9.

 topography e Overview for 33 years in Miyazaki Japan, Please cite this article in press as: Kuroki H, et al., School scoliosis screening by Moire Journal of Orthopaedic Science (2018), https://doi.org/10.1016/j.jos.2018.03.005