Assessment of the respiratory system in children with congenital scoliosis by impulse oscillometry and computed tomography (preliminary results)

Abstract

BACKGROUND: Segmentation disorder of the vertebral body lateral surfaces and rib synostosis are severe variants of congenital pathology of the spine and thorax. They lead to the development of thoracic insufficiency syndrome and are manifested by the inability of the thorax to provide normal respiratory mechanics.

AIM: This study presents the preliminary results of functional and radiological (CT-morphometric) methods of lung examinations in patients with congenital thoracic spine scoliosis with impaired segmentation of the lateral surfaces of the vertebral bodies and unilateral rib synostosis.

MATERIALS AND METHODS: This design is represented by a small clinical series. This study is a prospective study of 10 patients aged 3 to 7 years with congenital spinal deformity, with impaired segmentation of the lateral surfaces of vertebral bodies and unilateral rib synostosis. This paper presents the preliminary results of the pulmonary function assessment by pulse oscillometry and CT morphometry in a 3D reconstruction of multispiral computer tomography (MSCT) of the thorax.

RESULTS: The study of respiratory function using pulse oscillometry revealed no respiratory impairment in seven observations, also reflected in the CT morphometry results. According to the Institute of Medicine (IOM), three children with detected ventilatory abnormalities showed the following parameters with the most significant changes: total respiratory impedance, resonance frequency, and frequency dependence of the resistive component. In all patients, the morphometric indexes of the lung scoring revealed during 3D modeling of the lung were completely consistent with the results of the lung function study by the IOM method.

CONCLUSIONS: Further study of the problem of respiratory function assessment in children with congenital scoliosis seems promising in diagnostic terms and for evaluating effective surgical treatment.

Keywords

segmentation disorder / congenital scoliosis / thoracic insufficiency syndrome / pulse oscillometry

References

[1] McMaster MJ, McMaster ME. Prognosis for congenital scoliosis due to a unilateral failure of vertebral segmentation. J Bone Joint Surg Am. 2013;95(11):972–979. DOI: 10.2106/JBJS.L.01096

[2] McMaster M.J., McMaster M.E. Prognosis for congenital scoliosis due to a unilateral failure of vertebral segmentation // J. Bone Joint Surg. Am. 2013. Vol. 95. No. 11. P. 972–979. DOI: 10.2106/JBJS.L.01096

[3] Winter RB. Congenital thoracic scoliosis with unilateral unsegmented bar, convex hemivertebrae, and fused concave ribs with severe progression after posterior fusion at age 2. Spine. 2012;37(8):E507–E510. DOI: 10.1097/BRS.0b013e31824ac401

[4] Winter R.B. Congenital thoracic scoliosis with unilateral unsegmented bar, convex hemivertebrae, and fused concave ribs with severe progression after posterior fusion at age 2 // Spine. 2021. Vol. 37. No. 8. P. E507–E510. DOI: 10.1097/BRS.0b013e31824ac401

[5] Mihajlovskij MV, Suzdalov VA. Sindrom torakal’noj nedostatochnosti pri infantil’nom vrozhdennom skolioze. Hirurgija pozvonochnika. 2010;(3):20–28. (In Russ.). DOI: 10.14531/ss2010.3.20-28

[6] Михайловский М.В., Суздалов В.А. Синдром торакальной недостаточности при инфантильном врожденном сколиозе // Хирургия позвоночника. 2010. № 3. С. 20–28. DOI: 10.14531/ss2010.3.20-28

[7] Campbell RM Jr, Smith MD. Thoracic insufficiency syndrome and exotic scoliosis. J Bone Joint Surg Am. 2007;89(Suppl 1):108–122. DOI: 10.2106/JBJS.F.00270

[8] Campbell R.M. Jr., Smith M.D. Thoracic insufficiency syndrome and exotic scoliosis // J. Bone Joint Surg. Am. 2007. Vol. 89. Suppl. 1. P. 108–122. DOI: 10.2106/JBJS.F.00270

[9] Mayer O, Campbell R, Cahill P, Redding G. Thoracic insufficiency syndrome. Curr Probl Pediatr Adolesc Health Care. 2016;46(3):72–97. DOI: 10.1016/j.cppeds.2015.11.001

[10] Mayer O., Campbell R., Cahill P., Redding G. Thoracic insufficiency syndrome // Curr. Probl. Pediatr. Adolesc. Health Care. 2016. Vol. 46. No. 3. P. 72–97. DOI: 10.1016/j.cppeds.2015.11.001

[11] Tong Y, Udupa JK, McDonough JM, et al. Quantitative dynamic thoracic MRI: Application to thoracic insufficiency syndrome in pediatric patients. Radiology. 2019;292(1):206–213. DOI: 10.1148/radiol.2019181731

[12] Tong Y., Udupa J.K., McDonough J.M. et al. Quantitative dynamic thoracic MRI: Application to thoracic insufficiency syndrome in pediatric patients // Radiology. 2019. Vol. 292. No. 1. P. 206–213. DOI: 10.1148/radiol.2019181731

[13] Vissarionov SV, Husainov NO, Kokushin DN. Analiz rezul’tatov hirurgicheskogo lechenija detej s mnozhestvennymi anomalijami razvitija pozvonkov i grudnoj kletki s ispol’zovaniem vnepozvonochnyh metallokonstrukcij. Ortopedija, travmatologija i vosstanovitel’naja hirurgija detskogo vozrasta. 2017;5(2):5–12. (In Russ.). DOI: 10.17816/PTORS525

[14] Виссарионов С.В., Хусаинов Н.О., Кокушин Д.Н. Анализ результатов хирургического лечения детей с множественными аномалиями развития позвонков и грудной клетки с использованием внепозвоночных металлоконструкций // Ортопедия, травматология и восстановительная хирургия детского возраста. 2017. Т. 5. № 2. C. 5–12. DOI: 10.17816/PTORS525

[15] Schlösser TPC, Kruyt MC, Tsirikos AI. Surgical management of early-onset scoliosis: indications and currently available techniques. Orthop Trauma. 2021;35(6):1877–1327. DOI: 10.1016/j.mporth.2021.09.004

[16] Schlösser T.P.C., Kruyt M.C., Tsirikos A.I. Surgical management of early-onset scoliosis: indications and currently available techniques // Orthop. Trauma. 2021. Vol. 35. No. 6. P. 1877–1327. DOI: 10.1016/j.mporth.2021.09.004

[17] Campbell RM Jr, Smith MD. Thoracic insufficiency syndrome and exotic scoliosis. J Bone Joint Surg Am. 2007;89(Suppl 1):108–122. DOI: 10.2106/JBJS.F.00270

[18] Campbell R.M. Jr., Smith M.D. Thoracic insufficiency syndrome and exotic scoliosis // J. Bone Joint Surg. Am. 2007. Vol. 89. Suppl. 1. P. 108–122. DOI: 10.2106/JBJS.F.00270

[19] Mayer O, Campbell R, Cahill P, Redding G. Thoracic insufficiency syndrome. Curr Probl Pediatr Adolesc Health Care. 2016;46(3):72–97. DOI: 10.1016/j.cppeds.2015.11.001

[20] Mayer O., Campbell R., Cahill P., Redding G. Thoracic insufficiency syndrome // Curr. Probl. Pediatr. Adolesc. Health Care. 2016. Vol. 46. No. 3. P. 72–97. DOI: 10.1016/j.cppeds.2015.11.001

[21] Campbell RM Jr, Smith MD, Mayes TC, et al. The characteristics of thoracic insufficiency syndrome associated with fused ribs and congenital scoliosis. J Bone Joint Surg Am. 2003;85(3):399–408. DOI: 10.2106/00004623-200303000-00001

[22] Campbell R.M. Jr., Smith M.D., Mayes T.C. et al. The characteristics of thoracic insufficiency syndrome associated with fused ribs and congenital scoliosis // J. Bone Joint Surg. Am. 2003. Vol. 85. No. 3. P. 399–408. DOI: 10.2106/00004623-200303000-00001

[23] Romberg K, Fagevik Olsén M, Kjellby-Wendt G, Lofdahl Hallerman K, Danielsson A. Thoracic mobility and its relation to pulmonary function and rib-cage deformity in patients with early onset idiopathic scoliosis: a long-term follow-up. Spine Deform. 2020;8(2):257–268. DOI: 10.1007/s43390-019-00018-y

[24] Romberg K., Fagevik Olsén M., Kjellby-Wendt G. et al. Thoracic mobility and its relation to pulmonary function and rib-cage deformity in patients with early onset idiopathic scoliosis: a long-term follow-up // Spine Deform. 2020. Vol. 8. No. 2. P. 257–268. DOI: 10.1007/s43390-019-00018-y

[25] Farrell J, Garrido E. Predicting preoperative pulmonary function in patients with thoracic adolescent idiopathic scoliosis from spinal and thoracic radiographic parameters. Eur Spine J. 2021;30(3):634–644. DOI: 10.1007/s00586-020-06552-y

[26] Farrell J., Garrido E. Predicting preoperative pulmonary function in patients with thoracic adolescent idiopathic scoliosis from spinal and thoracic radiographic parameters // Eur. Spine J. 2021. Vol. 30. No. 3. P. 634–644. DOI: 10.1007/s00586-020-06552-y

[27] Hedequist DJ. Surgical treatment of congenital scoliosis. Orthop Clin North Am. 2007;38(4):497–509. DOI: 10.1016/j.ocl.2007.05.002

[28] Hedequist D.J. Surgical treatment of congenital scoliosis // Orthop. Clin. North Am. 2007. Vol. 38. No. 4. P. 497–509. DOI: 10.1016/j.ocl.2007.05.002

[29] Campos MA, Weinstein SL. Pediatric scoliosis and kyphosis. Neurosurg Clin N Am. 2007;18(3):515–529. DOI: 10.1016/j.nec.2007.04.007

[30] Campos M.A., Weinstein S.L. Pediatric scoliosis and kyphosis // Neurosurg. Clin. N. Am. 2007. Vol. 18. No. 3. P. 515–529. DOI: 10.1016/j.nec.2007.04.007

[31] Davydova IV, Namazova-Baranova LS, Altunin VV, et al. Functional assessment of respiratoty disorders in children with bronchopulmonary dysplasia during follow-up. Pediatricheskaja farmakologija. 2014;11(6):42–51. (In Russ.). DOI: 10.15690/pf.v11i6.1214

[32] Давыдова И.В., Намазова-Баранова Л.С., Алтунин В.В. и др. Функциональная оценка респираторных нарушений у детей с бронхолегочной дисплазией при катамнестическом наблюдении // Педиатрическая фармакология. 2014. Т. 11. № 6. С. 42–51. DOI: 10.15690/pf.v11i6.1214

[33] Yashina LA, Polyanskaya MA, Zagrebel’nyy M.R. Impul’snaya ostsillometriya – novye vozmozhnosti v diagnostike i monitoringe obstruktivnykh zabolevaniy legkikh. Zdorov’ya Ukraїni. 2009;(23/1):26−27. (In Russ.)

[34] Яшина Л.А., Полянская М.А., Загребельный М.Р. Импульсная осциллометрия – новые возможности в диагностике и мониторинге обструктивных заболеваний легких // Здоров’я України. 2009. № 23/1. С. 26−27.

[35] Cyplenkova SJe, Mizernickij JuL. Sovremennye vozmozhnosti funkcional’noi diagnostiki vneshnego dyhaniya u detei. Ros vestn perinatol i pediat. 2015;60(5):14−20. (In Russ.)

[36] Цыпленкова С.Э., Мизерницкий Ю.Л. Современные возможности функциональной диагностики внешнего дыхания у детей // Российский вестник перинатологии и педиатрии. 2015. Т. 60. № 5. С. 14−20.

[37] Feldman DS, Schachter AK, Alfonso D, et al. Congenital scoliosis. Surg Manag Spinal Deformities. 2009:129–141. DOI: 10.1016/B978-141603372-1.50012-3

[38] Feldman D.S., Schachter A.K., Alfonso D. et al. Congenital scoliosis // Surg. Manag. Spinal Deformities. 2009. P. 129–141. DOI: 10.1016/B978-141603372-1.50012-3

[39] Quaye M, Harvey J. Introduction to spinal pathologies and clinical problems of the spine. Biomaterials for Spinal Surgery. 2012:78–113. DOI: 10.1533/9780857096197.1.78

[40] Quaye M., Harvey J. Introduction to spinal pathologies and clinical problems of the spine // Biomaterials for Spinal Surgery. 2012. P. 78–113. DOI: 10.1533/9780857096197.1.78

[41] Blevins K, Battenberg A, Beck A. Management of scoliosis. Advances in Pediatrics. 2018;65(1):249–266. DOI: 10.1016/j.yapd.2018.04.013

[42] Blevins K., Battenberg A., Beck A. Management of scoliosis // Advances in Pediatrics. 2018. Vol. 65. No. 1. P. 249–266. DOI: 10.1016/j.yapd.2018.04.013

[43] Desai U, Joshi JM. Impulse oscillometry. Adv Respir Med. 2019;87(4):235–238. DOI: 10.5603/ARM.a2019.0039

[44] Desai U., Joshi J.M. Impulse oscillometry // Adv. Respir. Med. 2019. Vol. 87. No. 4. P. 235–238. DOI: 10.5603/ARM.a2019.0039

[45] Savushkina OI, Chernyak AV, Kryukov EV, et al. Impul’snaya ostsillometriya v diagnostike narusheniy mekhaniki dykhaniya pri khronicheskoy obstruktivnoy bolezni legkikh. Pul’monologiya. 2020;30(3):285–294. (In Russ.). DOI: 10.18093/0869-0189-2020-30-3-285-294

[46] Савушкина О.И., Черняк А.В., Крюков Е.В. и др. Импульсная осциллометрия в диагностике нарушений механики дыхания при хронической обструктивной болезни легких // Пульмонология. 2020. Т. 30. № 3. С. 285–294. DOI: 10.18093/0869-0189-2020-30-3-285-294

[47] Antonova E.A. Diagnostika narusheniy vneshnego dykhaniya u detey mladshego vozrasta (3–7 let), bol’nykh bronkhial’noy astmoy, po dannym impul’snoy ostsillometrii. Saint Petersburg; 2004. (In Russ.)

[48] Антонова Е.А. Диагностика нарушений внешнего дыхания у детей младшего возраста (3–7 лет), больных бронхиальной астмой, по данным импульсной осциллометрии: дис. … канд. мед. наук. Санкт-Петербург, 2004.

[49] Redding G, Song K, Inscore S, et al. Lung function asymmetry in children with congenital and infantile scoliosis. Spine J. 2008;8(4):639–644. DOI: 10.1016/j.spinee.2007.04.020

[50] Redding G., Song K., Inscore S. et al. Lung function asymmetry in children with congenital and infantile scoliosis // Spine J. 2008. Vol. 8. No. 4. P. 639–644. DOI: 10.1016/j.spinee.2007.04.020

[51] Flesch JD, Dine CJ. Lung volumes: measurement, clinical use, and coding. Chest. 2012;142(2):506–510. DOI: 10.1378/chest.11-2964

[52] Flesch J.D., Dine C.J. Lung volumes: measurement, clinical use, and coding // Chest. 2012. Vol. 142. No. 2. P. 506–510. DOI: 10.1378/chest.11-2964

[53] Caliskan E, Ozturk M. Determination of normal lung volume using computed tomography in children and adolescents. Original Article. 2019;26(4):588–592. DOI: 10.5455/annalsmedres.2018.12.308

[54] Caliskan E., Ozturk M. Determination of normal lung volume using computed tomography in children and adolescents // Original Article. 2019. Vol. 26. No. 4. P. 588–592. DOI: 10.5455/annalsmedres.2018.12.308

[55] Lattig F, Taurman R, Hell AK. Treatment of early-onset spinal deformity (EOSD) with VEPTR. Clinical Spine Surg. 2016;29(5):E246–E251. DOI: 10.1097/BSD.0b013e31826eaf27

[56] Lattig F., Taurman R., Hell A.K. Treatment of early-onset spinal deformity (EOSD) with VEPTR // Clinical Spine Surg. 2016. Vol. 29. No. 5. P. E246–E251. DOI: 10.1097/BSD.0b013e31826eaf27

[57] Li C, Fu Q, Zhou Y, et al. Surgical treatment of severe congenital scoliosis with unilateral unsegmented bar by concave costovertebral joint release and both-ends wedge osteotomy via posterior approach. European Spine Journal. 2011;21(3):498–505. DOI: 10.1007/s00586-011-1972-6

[58] Li C., Fu Q., Zhou Y. et al. Surgical treatment of severe congenital scoliosis with unilateral unsegmented bar by concave costovertebral joint release and both-ends wedge osteotomy via posterior approach // European Spine Journal. 2011. Vol. 21. No. 3. P. 498–505. DOI: 10.1007/s00586-011-1972-6

[59] Fender D, Purushothaman B. Spinal disorders in childhood II: spinal deformity. Surgery (Oxford). 2014;32(1):39–45. DOI: 10.1016/j.mpsur.2013.11.001

[60] Fender D., Purushothaman B. Spinal disorders in childhood II: spinal deformity // Surgery (Oxford). 2014. Vol. 32. No. 1. P. 39–45. DOI: 10.1016/j.mpsur.2013.11.001

[61] Campbell RM. Operative strategies for thoracic insufficiency syndrome by vertical expandable prosthetic titanium rib expansion thoracoplasty. Operative Techniques in Orthopaedics. 2005;15(4):315–325. DOI: 10.1053/j.oto.2005.08.008

[62] Campbell R.M. Operative strategies for thoracic insufficiency syndrome by vertical expandable prosthetic titanium rib expansion thoracoplasty // Operative Techniques in Orthopaedics. 2005. Vol. 15. No. 4. P. 315–325. DOI: 10.1053/j.oto.2005.08.008

[63] Loughenbury PR, Gummerson NW, Tsirikos AI. Congenital spinal deformity: assessment, natural history and treatment. Orthop Trauma. 2017;31(6):364–369. DOI: 10.1016/j.mporth.2017.09.007

[64] Loughenbury P.R., Gummerson N.W., Tsirikos A.I. Congenital spinal deformity: assessment, natural history and treatment // Orthop. Trauma. 2017. Vol. 31. No. 6. P. 364–369. DOI: 10.1016/j.mporth.2017.09.007

[65] Kalidindi KKV, Sath S, Sharma J, Chhabra HS. Management of severe rigid scoliosis by total awake correction utilizing differential distraction and in situ stabilization. Interdisciplinary Neurosurgery. 2020;21:100778. DOI: 10.1016/j.inat.2020.100778

[66] Kalidindi K.K.V., Sath S., Sharma J., Chhabra H.S. Management of severe rigid scoliosis by total awake correction utilizing differential distraction and in situ stabilization // Interdisciplinary Neurosurgery. 2020. Vol. 21. P. 100778. DOI: 10.1016/j.inat.2020.100778

[67] Campbell R, Hell-Vocke AK. The growth of the thoracic spine in congenital scoliosis after expansion thoracoplasty. Spine. 2002;2(5 Suppl):71–72. DOI: 10.1016/S1529-9430(02)00317-0

[68] Campbell R., Hell-Vocke A.K. The growth of the thoracic spine in congenital scoliosis after expansion thoracoplasty // Spine. 2002. Vol. 2. No. 5. Suppl. P. 71–72. DOI: 10.1016/S1529-9430(02)00317-0

[69] Skaggs, DL, Guillaume T, El-Hawary R, et al. (2015). Early onset scoliosis consensus statement, SRS Growing Spine Committee, 2015. Spine Deformity. 2015;3(2):107. DOI: 10.1016/j.jspd.2015.01.002

[70] Skaggs D.L., Guillaume T., El-Hawary R. et al. Early onset scoliosis consensus statement, SRS Growing Spine Committee, 2015 // Spine Deformity. 2015. Vol. 3. No. 2. P. 107. DOI: 10.1016/j.jspd.2015.01.002

[71] Lukina O.F. Osobennosti issledovaniya funktsii vneshnego dykhaniya u detey i podrostkov. Prakticheskaya pul’monologiya. 2017(4):39–44. (In Russ.)

[72] Лукина О.Ф. Особенности исследования функции внешнего дыхания у детей и подростков // Практическая пульмонология. 2017. № 4. С. 39–44.

[73] Lattig F, Taurman R, Hell AK. Treatment of early-onset spinal deformity (EOSD) with VEPTR. Clinical Spine Surgery. 2016;29(5):E246–E251. DOI: 10.1097/BSD.0b013e31826eaf27

[74] Lattig F., Taurman R., Hell A.K. Treatment of early-onset spinal deformity (EOSD) with VEPTR // Clinical Spine Surgery. 2016. Vol. 29. No. 5. P. E246–E251. DOI: 10.1097/BSD.0b013e31826eaf27

[75] Lonstein JE. Long-term outcome of early fusions for congenital scoliosis. Spine Deformity. 2018;6(5):552–559. DOI: 10.1016/j.jspd.2018.02.003

[76] Lonstein J.E. Long-term outcome of early fusions for congenital scoliosis // Spine Deformity. 2018. Vol. 6. No. 5. P. 552–559. DOI: 10.1016/j.jspd.2018.02.003

[77] Murphy RF, Pacult MA, Barfield WR, et al. Experience with definitive instrumented final fusion after posterior-based distraction lengthening in patients with early-onset spinal deformity. J Pediatr Orthop B. 2019;28(1):10–16. DOI: 10.1097/BPB.0000000000000559

[78] Murphy R.F., Pacult M.A., Barfield W.R. et al. Experience with definitive instrumented final fusion after posterior-based distraction lengthening in patients with early-onset spinal deformity: single center results // J. Pediatr. Orthop. B. 2019. Vol. 28. No. 1. P. 10–16. DOI: 10.1097/BPB.0000000000000559

[79] Johnston CE, Stephens Richards B, Sucato DJ, et al. Correlation of preoperative deformity magnitude and pulmonary function tests in adolescent idiopathic scoliosis. Spine. 2011;36:1096–1102. DOI: 10.1097/BRS.0b013e3181f8c931

[80] Johnston C.E., Stephens Richards B., Sucato D.J. et al. Correlation of preoperative deformity magnitude and pulmonary function tests in adolescent idiopathic scoliosis // Spine. 2011. Vol. 36. P. 1096–1102. DOI: 10.1097/BRS.0b013e3181f8c931

[81] Karol LA. The natural history of early-onset scoliosis. J Pediatr Orthop. 2019;39(6 Suppl 1):S38–S43. DOI: 10.1097/BPO.0000000000001351

[82] Karol L.A. The natural history of early-onset scoliosis // J. Pediatr. Orthop. 2019. Vol. 39. No. 6. Suppl. 1. P. S38–S43. DOI: 10.1097/BPO.0000000000001351

[83] Tomlinson JE, Gummerson NW. Paediatric spinal conditions. Surgery (Oxford). 2017;35(1):39–47. DOI: 10.1016/j.mpsur.2016.10.013

[84] Tomlinson J.E., Gummerson N.W. Paediatric spinal conditions // Surgery (Oxford). 2017. Vol. 35. No. 1. P. 39–47. DOI: 10.1016/j.mpsur.2016.10.013

[85] Gardner A. (i) Clinical assessment of scoliosis. Orthop Trauma. 2011;25(6):397–402. DOI: 10.1016/j.mporth.2011.09.002

[86] Gardner A. (i) Clinical assessment of scoliosis // Orthop. Trauma. 2011. Vol. 25. No. 6. P. 397–402. DOI: 10.1016/j.mporth.2011.09.002

[87] Duenas-Meza E, Correa E, Lopez E, et al. Impulse oscillometry reference values and bronchodilator response in three-to five-year old children living at high altitude. J Asthma Allergy. 2019;12:263–271. DOI: 10.2147/JAA.S214297

[88] Duenas-Meza E., Correa E., Lopez E. et al. Impulse oscillometry reference values and bronchodilator response in three- to five-year old children living at high altitude // J. Asthma Allergy. 2019. Vol. 12. P. 263–271. DOI: 10.2147/JAA.S214297

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Vissarionov S.V., Asadulaev M.S., Orlova E.A., Toriya V.G., Kartavenko K.A., Rybinskikh T.S., Murashko T.V., Khardikov M.A., Kokushin D.N.