117 pissn : , eissn : Symposium J Korean Orthop Assoc 2016; 51: C

117 pissn : 1226-2102, eissn : 2005-8918 Symposium J Korean Orthop Assoc 2016; 51: 117-124 http://dx.doi.org/10.4055/jkoa.2016.51.2.117 www.jkoa.org Common Problems in Skeletally Immature Patients 유소년형특발성척추측만증 강민석 서승우 * 최승진 황진호 건국대학교의학전문대학원정형외과학교실, * 고려대학교의과대학정형외과학교실, 연세대학교의과대학정형외과학교실 Juvenile Idiopathic Scoliosis Min Seok Kang, M.D., Seung Woo Suh, M.D.*, Seungjin Choi, M.D., and Jin-Ho Hwang, M.D. Department of Orthopedic Surgery, Konkuk University School of Medicine, *Department of Orthopedic Surgery, Korea University School of Medicine, Department of Orthopedic Surgery, Yonsei University School of Medicine, Seoul, Korea Juvenile idiopathic scoliosis includes scoliosis diagnosed from three to ten years old according to the chronological age. Spine growth in juveniles does not occur at a rapid rate spinal deformity does not show rapid progress. However, because of the intimate relationship between chest wall growth and the spine, decrease of chest wall capacity due to scoliosis could lead to development of cardiovascular and pulmonary complication, especially in early age. In scoliosis in early age, other causes of the deformity including neurological problems should be evaluated. If the scoliosis angle is more than 25 degrees, it could progress very easily, thus aggressive treatment is needed. A new growing-sparing surgical technique (growing rod and growth modulation) is introduced for improvement of spine and chest growth, and for prevention of crankshaft phenomenon. Key words: juvenile idiopathic scoliosis, spinal growth, chest growth, growing rod, growth modulation 서론 특발성척추측만증은진단된때의역연령 (chronological age) 에따라분류한다. 유아기형 (infantile idiopathic scoliosis, 유아기형측만증 ) 은태생후 3세까지, 유소년형 ( 유소년기형측만증 ) 은 3세이후 10세미만까지, 청소년형은 10세이후에발병된경우이다. 유소년기의척추의성장은빠르지않는특징을가지고있기때문에모든척추변형이빠르게진행하지않으며, 이에따른특징을갖는다. 1,2) 척추측만증은성장과밀접한관계를가지며진행하기때문에유소년기형측만증에대한적절한치료를위해서는정상척추의발달과성장및특징을이해하고알아야한다. Received July 28, 2015 Revised vember 30, 2015 Accepted February 3, 2016 Correspondence to: Jin-Ho Hwang, M.D. Department of Orthopedic Surgery, Yonsei University School of Medicine, 50-1 Yonsei-ro, Seodaemun-gu, Seoul 03722, Korea TEL: +82-2-2228-2180 FAX: +82-2-363-1139 E-mail: osjinho@naver.com 척추와흉곽의성장과발달 Dimeglio와 Canavese 3) 는태생후 5세까지평균척추성장속도는 1년에 2 cm 이상으로가장빠르고, 6-10세사이는 1년에 0.5 cm로매우둔화되며, 11-18세사이에이차급성장기와더불어 1.3 cm/ yr로다시증가한다고하였다 (Fig. 1). 2,4) 흉곽의성장 (chest wall growth) 은척추의성장과밀접한관계가있어서척추의길이가짧아지면흉곽의용적은매우감소한다. 흉곽은태생시성인의 5% 이나 5세까지 30% 까지 6배이상성장한다. 이후둔화되어 10세에 50%, 15세에성인의흉곽용적에도달한다. 따라서조기발현형일수록흉곽용적의감소가두드러지고이로인한심혈관및호흡기적합병증의발생이높아진다 (Fig. 2). 자연경과및역학 특발성척추측만증중유소년기형측만증이차지하는비율은 The Journal of the Korean Orthopaedic Association Volume 51 Number 2 2016 Copyright 2016 by The Korean Orthopaedic Association This is an Open Access article distributed under the terms of the Creative Commons Attribution n-commercial License (http://creativecommons.org/licenses/by-nc/4.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

118 Min Seok Kang, et al. 12%-21% 정도이다. 5) 남녀성비에따른유병률은점진적으로나 이가증가함에따라청소년기형측만증과유사해진다. 3-6세에서측만증발생의남녀의비는 1:1이고나이가증가함에따라 1:2-1:4 의비율로증가한다. 10세에가까워지면서 1:8 의비율로청소년기측만증과유사해진다 6) 대부분남아는 5세경, 여아는 7 세경에진단된다. 유소년기형측만증의자연경과는유아기형측만증과청소년기형측만증의 이행기적특징 (gradual transition) 을갖는다. 일찍발병하면빠르게진행하며심한변형이남아수술이필요한경우가많다. 그러나전반적으로는 slow-to moderate progression 하 cm/yr 2.5 1.5 1.0 0.5 0 Early onset Late onset 0.25 1 5 10 15 Age (yr) T1-L5 T1-T12 L1-L5 1.75 Figure 1. Spinal growth from ages 1 through 15 years. Spinal growth decelerates during the juvenile period, between the age of 5 and 10 years. There is a second acceleration of the growth rate along with the puberty in the adolescent period. Therefore, early onset scoliosis is often defined as scoliosis that manifests before the age of 5 years, as compared to late onset scoliosis which manifests after the age of 5 years. 0.5 는것으로알려져있다. 5-8) 유소년기형측만증중 70% 는진행하며대부분치료를필요로하는것으로보고된다. Tolo와 Gillespie 9) 는 59명중 42명 (71.2%) 의환자가수술적치료를필요로하였다고보고하였고, Figueiredo와 James 8) 는 98명중 55명 (56.1%) 에서수술적치료를요하였다고하였다. 그러나 Mannherz 등 10) 은 25도미만의경미한경우에서진행하 cm/yr 2.5 1.5 1.0 0.5 0 6.4% New born 14 cm/yr 0.6 cm/yr 0.5 1.6 cm/yr 0.25 1.75 1 5 10 15 Age (yr) 100% 50% 30% Figure 2. Growth of spine and thorax. The thoracic volume is 5% adulthood at birth, which increases to more than 30% at the age of 5 years. Early onset of scoliosis relates to reduced spinal length, which can ultimately lead to decreased thoracic volume and increased risk of pulmonary complications. 1.0 T1-L5 T1-T12 L1-L5 T11 30 15 L3 Figure 3. Serial radiographs of a 7-yearold boy with an initial deformity curve of 30 degrees. A 1 month follow-up radiograph showed resolution of the curve. After 2 months, a curve measuring 15 degrees is seen again in the radiograph. This case demonstrates the features of juvenile idiopathic scoliosis well.

119 Juvenile Idiopathic Scoliosis 지않고저절로회복되는경우를보고하였다. Modi 등 11) 은 169 명 의환아에서진행한경우는 26% 뿐이며 32.5% 의경우는자연회복되었고, 나머지 41.4% 에서는측만증의형태가지속적으로변화되는과정에있다고보고하여 tuning/balancing mechanism 의가설을제시하였다 (Fig. 3, 4). 임상적평가 어린나이에측만증이발병하는경우다른원인이동반되는경우가많으며, 특히청소년기형측만증과달리유소년기형측만증에서는신경이상 (intra-spinal abnormality) 이동반될가능성이매우높다. 동반된원인을제거하면측만증의진행을느리게하거나정지시킬수있다. 특발성척추측만증은정의상척추변형을유발하는다른원인들이배제되고원인이밝혀지지않은경우를의미하기때문에신경근육성척추측만증 (neuromuscular scoliosis), 척수공동증, 척추종양, 선청성척추측만증, 신경섬유종, 여러신드롬에동반되어나타나는척추측만증 (syndromic scoliosis) 등다른원인에의한척추측만증들을배제하여야한다. Cobb angle Spinal balancing period After spinal balancing phase x y z Figure 4. Tuning/balancing mechanism. Tuning/balancing mechanism is a characteristic feature of juvenile idiopathic scoliosis which the Cobb angle shows variation over time. The process of progression, quit and regression can be seen. Figure shows x is the onset time of scoliosis in growing spine; y is the time when curve will follow one of three pathway (A: regression; B: quit and C: progression); and z is the time of change in treatment approach. C B A 특히척수공동증과동반된 Chiari malformation에서척추측만증의발생률은 20% 로보고되기도하므로주의를요하며, 동반증상으로경미한두통및경부통증이있을수있다 (Table 1). 12-17) 유소년기형측만증환아의 20% 정도에서자기공명영상촬영상신경이상이동반된다. 이는청소년기형측만증에비해높기때문에만곡이급속히진행하거나각도가 25도이상의척추측만에서추시상만곡의형태가변하지않는경우에는자기공명영상촬영을시행하는것이좋다 (Fig. 5). 18,19) 기왕력청취시심장기형, 고관절의이상, 인지능력저하, 선천성사경등도주의깊게관찰해야한다. 특히유소년기에성장호르몬치료 (growth hormone) 를시행하는경우에는세심한기왕력청취가필요하다. 성장호르몬은척추의성장을촉진시켜척추측만증을악화시킨다는보고도있으나성장호르몬을투여하지않은군과비교하여더높지않다는보고도있어 20-22) 성장호르몬치료를받는환아에서는측만증발병및진행가능성에대한충분한설명과주기적인추적관찰이필요하다할수있다. 근래에는키와관련하여하지부동 (lower limb discrepancy) 과척추측만증에대한관심도높아지고있다. 하지부동이반드시척추측만증을발생시키지는않으나내원시하지부동에대한평가를하는것이좋다. 깔창을이용한하지부동교정이측만증의교정이나소실에효과가없는것으로보고되고있으므로깔창으로무리한교정을하기보다는 교정신발 착용후방사선사진을찍고척추측만증이소실되는경우에 교정신발 을추천한다. 23,24) 치료 유소년기형측만증의치료는청소년기형측만증과같이명확히정립되어있지않다. 각도의크기, 환자의나이, 잔여성장, 만곡의유연성, 기저질환등을고려하여치료방침을결정하고, 만곡이 25도이상인경우에는진행의가능성이매우높기때문에적극적치료를요한다. Fig. 6은치료의방향을제시하는알고리즘이다. 보조기치료는청소년기형측만증과달리 60도이상에서도착용을시도해볼수있다. 25,26) 유소년기형측만증은청소년기형측만증에비해만곡이상당히유연하기때문이다. 또한최근에는 Table 1. Presenting Symptoms in the Syringomyelia and Chiari Malformation Author (yr) Total HA/neck pain Sensory loss Motor loss CN/BS/cerebellar Ataxia Scoliosis Navarro et al. (2004) 12) 96 53 (55.2) NS NS 19 (19.8) NS 14 (14.6) Tubbs et al. (2003) 13) 130 55 (42.3) NS NS NS 12 (9.2) 23 (17.7) Alzate et al. (2001) 14) 66 36 (54.5) 20 (30.3) 8 (12.1) 14 (21.2) 4 (6.1) 11 (16.7) Park et al. (1997) 15) 68 43 (63.2) 18 (26.5) 13 (19.1) NS NS 19 (27.9) Ellenbogen et al. (2000) 16) 36 27 (75.0) 20 (55.6) 17 (47.2) NS 9 (25.0) 19 (52.8) Krieger et al. (1999) 17) 31 16 (51.6) 9 (29.0) 12 (38.7) NS 7 (22.6) 20 (64.5) Values are presented as number only or number (%). Mild degree of neck pain take up a high percentage of the patient s symptom. Scoliosis can be the first presenting sign. HA, headache; CN/BS, cranial nerve/brain stem; NS, not stated.

120 Min Seok Kang, et al. T7 29 L1 A B C 34 Figure 5. (A) A 6 year and 9 monthold boy with a 29 degree of scoliosis in the radiograph. (B) Magnetic resonance imaging scan of the spine revealed syringomyelia (big arrow) with Chiari malformation (small arrow) in the cord and surgical treatment was performed. (C) After surgery, signs of syringomyelia has improved and the deformity angle remains stable with the conservative treatment of bracing. 수술법의발전으로만곡이 60도이상이어도교정수술이가능하고, 일찍수술을하면척추의성장이되지않기때문에가능하면척추의성장이완성될때까지보조기를이용한치료가권장된다. 유소년기측만증은척추가성장하는시기이므로수술적치료시다음과같은사항이고려된다. 첫째는척추유합술을시행하는경우척추의성장이정지되면서흉곽의성장이멈추어심혈관및호흡기계합병증을유발할수있다. 또한환아의키가줄어들어외관상의문제를만들수있다. Klemme 등 27) 은유합하는척추마디수에 0.07을곱하면 ( 척추마디유합수 0.07) 척추길이의손실을계산할수있다하였다. 둘째는척추의후방만유합하고전방척추를유합하지않을경우, 유합된후방척추는성장이되지않는반면에유합을하지않은척추의전방은성장되면서, 전방과후방척추성장의불균형으로척추가꽈배기모양으로꼬이는 crankshaft 현상이발생한다. 28) 이러한현상은성장이남아있는상태에서후방교정을시행한 경우나삼방연골 (triradiate cartilage) 이유합되지않은상태에서후방유합을시행한경우발생한다. 29) 현재치료의초점은척추자체만을보는것에서척추, 흉곽, 폐까지보는것으로변화되었다. 적절한폐의성장과기능을유지하기위한충분한흉곽과잘정렬된척추를만드는것이치료의목적이다. 이러한개념의수술방법을통창하여 성장유지수술법 (growing-sparing surgical technique) 이라한다. 수술의방법은 성장막대 (growing rod) 와 성장조율 (growth modulation) 의두가지방법이있다 (Table 2). 성장형막대를이용한수술법은척추를영구적으로고정하는것이아니고, 성장이가능한금속봉을사용하여척추의성장에따라 1년에 1-2회정도금속봉의길이를늘려주는 distraction-based 수술방법이다. 현재장기추시결과는없으나 Akbarnia 등 30) 은 2005년연구에서 dual-growing rod 를사용하여척추측만각이평

121 Juvenile Idiopathic Scoliosis JIS (5-10 years of age) Abnormal neurologic finding or Cobb angle of 25 or more? Comprehensive history and physical and scoliosis radiographs Significant nonorthopaedic finding MRI of spinal cord positive finding Specialty referral for nonorthopaedic conditions Neurosurgery specialty evaluation Continue with orthopaedic management Cobb angle of 25 or more? Casting/bracing Serial observation every 4-6 months Good response Progression of curve Annual clinical examination until skeletal maturity) Growing rod +/ traction or anterior release Serial lengthenings every 4-6 months Possible removal of instrumentation and continued observation Consider surgical intervention Definitive fusion Other emerging techniques Figure 6. Treatment algorithm of juvenile idiopathic scoliosis (JIS). MRI: magnetic resonance imaging. Table 2. Summary of the Surgical Techniques Used Recently Distraction based Guided growth Convex compression growth inhibition Other Single rod Luque trolley Shape memory staples Rib lengthening/shortening Dual rod Shilla Tethers Neurocentral synchodrosis hemiepiphyseodesis VEPTR Hybrid growth rods Phenix/MAGEC 균 82도에서 38도로교정되었으며최종 36도였다고보고하였다. 평균 6.6회의연장횟수로년간 1.2 cm를연장하였으며. 11명의환자에게서 13개의합병증 (56%) 을보고하였다. 합병증발생이높은것이이술기의문제이며 140명을분석한 대규모연구에서 58% 의환자에서합병증이발생하였고최소한번이상이었다고하였다. 31) 수술의횟수증가에따른상처감염이가장흔한합병증이며, 금속봉골절및신경이상등현재까지많은합병증이있어사용에세심한주의를요한다. 32,33) 근래에는이

122 Min Seok Kang, et al. 러한수술횟수증가와합병증의비례로인해수술의횟수를줄이기위한방안으로절개를시행하지않고원격조정하는방법이개발중이다 (Phenix/MAGEC). 34,35) 대표적성장조율방법은 Hueter-Volkman Law 를기반으로신전력 (tension) 에의해성장이촉진되어있는변형의볼록부위에압박력 (compression) 을가해성장을저하시키는것이다. 이는소아에서의골단고정술 (epiphysiodesis) 의방법과동일하다. 성장이촉진되어있는척추측만증의볼록부위에수술을하여성장을저하시킴으로써진행을막고교정을시도하는방법이다. 이방법은처음에선천성척추측만증에처음시도되었다. 36-39) 최근에는 형상기억합금 (shape memory alloy, SMA) staple, 40-43) tethers 44-46) 등많은방법이시도되고있다. Betz 등 47) 은 SMA의일종인 Nitinol (Nickel Titanium Naval Ordance Lab) 을사용하여평균나이 10세미만의 25명의흉부만곡과 15명의요추만곡에대해수술을시행하였다. 흉부만곡에대해서 35도미만에서 78% 에서호전, 50도이상에서는 75% 에서진행한다하였으며 10세미만에서는 75% 의성공률을보인다고보고하였다. 요추만곡은전체적으로 87% 의성공률과 10세미만의경우 100% 의성공률을보고하였다. 따라서 45도미만의 20도이상의유연성을가진경우적응증이라하였다. CONFLICTS OF INTEREST The authors have nothing to disclose. REFERENCES 1. Pehrsson K, Larsson S, Oden A, Nachemson A. Long-term follow-up of patients with untreated scoliosis. A study of mortality, causes of death, and symptoms. Spine (Phila Pa 1976). 1992;17:1091-6. 2. Little DG, Sussman MD. The Risser sign: a critical analysis. J Pediatr Orthop. 1994;14:569-75. 3. Dimeglio A, Canavese F. The growing spine: how spinal deformities influence normal spine and thoracic cage growth. Eur Spine J. 2012;21:64-70. 4. Lonstein JE, Carlson JM. The prediction of curve progression in untreated idiopathic scoliosis during growth. J Bone Joint Surg Am. 1984;66:1061-71. 5. Ponseti IV, Friedman B. Prognosis in idiopathic scoliosis. J Bone Joint Surg Am. 1950;32:381-95. 6. James JI. Idiopathic scoliosis; the prognosis, diagnosis, and operative indications related to curve patterns and the age at onset. J Bone Joint Surg Br. 1954;36:36-49. 7. Koop SE. Infantile and juvenile idiopathic scoliosis. Orthop Clin rth Am. 1988;19:331-7. 8. Figueiredo UM, James JI. Juvenile idiopathic scoliosis. J Bone Joint Surg Br. 1981;63:61-6. 9. Tolo VT, Gillespie R. The characteristics of juvenile idiopathic scoliosis and results of its treatment. J Bone Joint Surg Br. 1978;60:181-8. 10. Mannherz RE, Betz RR, Clancy M, Steel HH. Juvenile idiopathic scoliosis followed to skeletal maturity. Spine (Phila Pa 1976). 1988;13:1087-90. 11. Modi HN, Suh SW, Yang JH, Hong JY, Venkatesh K, Muzaffar N. Spontaneous regression of curve in immature idiopathic scoliosis - does spinal column play a role to balance? An observation with literature review. J Orthop Surg Res. 2010;5:80. 12. Navarro R, Olavarria G, Seshadri R, Gonzales-Portillo G, McLone DG, Tomita T. Surgical results of posterior fossa decompression for patients with Chiari I malformation. Childs Nerv Syst. 2004;20:349-56. 13. Tubbs RS, McGirt MJ, Oakes WJ. Surgical experience in 130 pediatric patients with Chiari I malformations. J Neurosurg. 2003;99:291-6. 14. Alzate JC, Kothbauer KF, Jallo GI, Epstein FJ. Treatment of Chiari I malformation in patients with and without syringomyelia: a consecutive series of 66 cases. Neurosurg Focus. 2001;11:E3. 15. Park JK, Gleason PL, Madsen JR, Goumnerova LC, Scott RM. Presentation and management of Chiari I malformation in children. Pediatr Neurosurg. 1997;26:190-6. 16. Ellenbogen RG, Armonda RA, Shaw DW, Winn HR. Toward a rational treatment of Chiari I malformation and syringomyelia. Neurosurg Focus. 2000;8:E6. 17. Krieger MD, McComb JG, Levy ML. Toward a simpler surgical management of Chiari I malformation in a pediatric population. Pediatr Neurosurg. 1999;30:113-21. 18. Gupta P, Lenke LG, Bridwell KH. Incidence of neural axis abnormalities in infantile and juvenile patients with spinal deformity. Is a magnetic resonance image screening necessary? Spine (Phila Pa 1976). 1998;23:206-10. 19. Lewonowski K, King JD, Nelson MD. Routine use of magnetic resonance imaging in idiopathic scoliosis patients less than eleven years of age. Spine (Phila Pa 1976). 1992;17:S109-16. 20. Bell J, Parker KL, Swinford RD, Hoffman AR, Maneatis T, Lippe B. Long-term safety of recombinant human growth hormone in children. J Clin Endocrinol Metab. 2010;95:167-77.

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124 pissn : 1226-2102, eissn : 2005-8918 Symposium J Korean Orthop Assoc 2016; 51: 117-124 http://dx.doi.org/10.4055/jkoa.2016.51.2.117 www.jkoa.org 성장기소아환자의흔한정형외과적문제 유소년형특발성척추측만증 강민석 서승우 * 최승진 황진호 건국대학교의학전문대학원정형외과학교실, * 고려대학교의과대학정형외과학교실, 연세대학교의과대학정형외과학교실 유소년형특발성척추측만증은역연령에따라 3세이후 10세미만까지의척추측만증을뜻한다. 유소년기의척추의성장은빠르지않기때문에척추변형이빠르게진행하지는않으나조기발현형일수록흉곽용적의감소가두드러지고이로인한심혈관및호흡기적합병증의발생이높아진다. 어린나이의측만증은신경이상등의척추변형을유발하는다른원인들이있는지를확인해야한다. 만곡이 25도이상인경우에는진행의가능성이매우높기때문에적극적치료를요한다. 수술의경우유합술은척추성장이정지되어흉곽의성장에영향을미치며, crankshaft 현상을막기위해전후방유합이필요하다. 최근에는 성장막대 (growing rod) 와 성장조율 (growth modulation) 의사용으로 성장유지수술법 (growing-sparing surgical technique) 이제시되고있다. 색인단어 : 유소년형특발성척추측만증, 척추성장, 흉곽성장, 성장막대, 성장조율 접수일 2015 년 7 월 28 일수정일 2015 년 11 월 30 일게재확정일 2016 년 2 월 3 일책임저자황진호 03722, 서울시서대문구연세로 50-1, 연세대학교의과대학정형외과학교실 TEL 02-2228-2180, FAX 02-363-1139, E-mail osjinho@naver.com 대한정형외과학회지 : 제 51권제 2호 2016 Copyright 2016 by The Korean Orthopaedic Association This is an Open Access article distributed under the terms of the Creative Commons Attribution n-commercial License (http://creativecommons.org/licenses/by-nc/4.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.