Abstract
Purpose
Kashin–Beck disease (KBD) is an endemic osteoarthropathy affecting the epiphyseal growth plate of multiple joints in young and adolescent patients. Previous studies have focused on the visible deformed extremities instead of the spinal radiological features, especially the atlantoaxial joint. The aim of this study was to determine the prevalence and radiographic features of atlantoaxial dislocation (AAD) in adult patients with KBD.
Methods
This study was conducted on KBD patients in three typical endemic counties between October 2017 and November 2019. The patients were evaluated by collecting basic information, clinical signs and symptoms. They underwent dynamic cervical radiography, by which AAD was diagnosed. For those patients with confirmed or suspected AAD, computed tomography (CT) imaging was performed to observe the odontoid morphology and degenerative changes in the lateral atlantoaxial joints. Radiographic evaluations were reviewed to determine the prevalence and features of AAD.
Results
A total of 39 (14.6%) of 267 KBD patients were diagnosed with AAD. Compared with the non-AAD patients, the detection rate of AAD was associated with a longer disease duration and stage and was not associated with age, sex or BMI. Thirty-two patients had symptoms at the neck or neurological manifestations, while seven had no symptoms. There were three types of morphologies of the odontoid process in AAD patients: separating in 19 cases, hypoplastic in 15 cases and intact in five cases. Anterior dislocation was noted in 29 cases, and posterior dislocation was noted in ten cases. Thirty-four cases were reducible, and five were irreducible. The lateral atlantoaxial joints had different severities of degenerative changes in 17 cases.
Conclusions
This study revealed that the prevalence of AAD was 14.6% in adult KBD patients. The radiographic features of AAD include manifestations of odontoid dysplasia and chronic degenerative changes in atlantoaxial joints. KBD patients with severe stages and longer disease duration were more vulnerable to the occurrence of AAD. We postulate that this atlantoaxial anomaly might originate from chondronecrosis of the epiphyseal growth plate of the odontoid process in young and adolescent individuals. This study may provide a clinical reference to help clinicians screen, prevent and treat AAD in adult patients with KBD.
Similar content being viewed by others
References
Guo X, Ma WJ, Zhang F, Ren FL, Qu CJ, Lammi MJ (2014) Recent advances in the research of an endemic osteochondropathy in China: Kashin-Beck disease. Osteoarthr Cartil 22(11):1774–1783. https://doi.org/10.1016/j.joca.2014.07.023
Hinsenkamp M (2001) Kashin-Beck disease. Int Orthop 25(3):133. https://doi.org/10.1007/s002640000177
National Health Commission of China (2021) China statistical year book of health and family planning. China Union Medical College Press, Beijing, p 272
Cao CX, Zhang YG, Wu SX, Younas MI, Guo X (2013) Association of clinical features of bone and joint lesions between children and parents with Kashin-Beck disease in Northwest China. Clin Rheumatol 32(9):1309–1316. https://doi.org/10.1007/s10067-013-2267-6
Schepman K, Engelbert RH, Visser MM, Yu C, de Vos R (2011) Kashin Beck Disease: more than just osteoarthrosis: a cross-sectional study regarding the influence of body function-structures and activities on level of participation. Int Orthop 35(5):767–776. https://doi.org/10.1007/s00264-010-1043-3
Xiong G (2001) Diagnostic, clinical and radiological characteristics of Kashin-Beck disease in Shaanxi Province, PR China. Int Orthop 25(3):147–150. https://doi.org/10.1007/s002640100248
Fu Q, Cao J, Renner JB, Jordan JM, Caterson B, Duance V, Luo M, Kraus VB (2015) Radiographic features of hand osteoarthritis in adult Kashin-Beck Disease (KBD): the Yongshou KBD study. Osteoarthr Cartil 23(6):868–873. https://doi.org/10.1016/j.joca.2015.01.009
Hinsenkamp M, Ryppens F, Begaux F, Mathieu F, De Maertelaer V, Lepeire M, Haubruge E, Chasseur C, Stallenberg B (2001) The anatomical distribution of radiological abnormalities in Kashin-Beck disease in Tibet. Int Orthop 25(3):142–146. https://doi.org/10.1007/s002640100236
Yang SY, Boniello AJ, Poorman CE, Chang AL, Wang S, Passias PG (2014) A review of the diagnosis and treatment of atlantoaxial dislocations. Global Spine J 4(3):197–210. https://doi.org/10.1055/s-0034-1376371
Yin QS, Wang JH (2015) Current trends in management of atlantoaxial dislocation. Orthop Surg 7(3):189–199. https://doi.org/10.1111/os.12196
Wu X, Ling M, Chang Y, Sun Z (2017) Accidental or linked: separated odontoid process fused to the enlarged anterior arch of the atlas associated with atlantoaxial subluxation in a Kashin-Beck disease patient. Eur Spine J 26(Suppl 1):85–89. https://doi.org/10.1007/s00586-016-4783-y
Kotecki M, Gasik R, Gluszko P, Sudol-Szopinska I (2021) Radiological evaluation of cervical spine involvement in rheumatoid arthritis: a cross-sectional retrospective study. J Clin Med 10(19):4587. https://doi.org/10.3390/jcm10194587
Yu FF, Ping ZG, Yao C, Wang ZW, Wang FQ, Guo X (2017) Evaluation of the sensitivity and specificity of the new clinical diagnostic and classification criteria for Kashin-Beck disease, an endemic osteoarthritis, in China. Biomed Environ Sci 30(2):150–155. https://doi.org/10.3967/bes2017.021
Ling M, Wu X, Chang Y, Dong X, Sun Z, Ling L, Wu S, Yang B (2017) Staged total knee arthroplasty for bilateral complex knee deformities from Kashin-Beck disease and skeletal dysplasia. Knee 24(3):692–698. https://doi.org/10.1016/j.knee.2016.11.011
Huang Q, Zhou ZK, Ma J, Li Y, Yang X, Shen B, Yang J, Kang PD, Pei FX (2015) The arthropathic and functional impairment features of adult Kashin-Beck disease patients in Aba Tibetan area in China. Osteoarthr Cartil 23(4):601–606. https://doi.org/10.1016/j.joca.2015.01.005
Chang H, Park JB, Kim KW, Choi WS (2000) Retro-dental reactive lesions related to development of myelopathy in patients with atlantoaxial instability secondary to Os odontoideum. Spine (Phila Pa 1976) 25(21):2777–2783. https://doi.org/10.1097/00007632-200011010-00010
Wang Q, Dong S, Wang F (2020) Os odontoideum: diagnosis and role of imaging. Surg Radiol Anat 42(2):155–160. https://doi.org/10.1007/s00276-019-02351-3
Jumah F, Alkhdour S, Mansour S, He P, Hroub A, Adeeb N, Hanif R, Mortazavi MM, Tubbs RS, Nanda A (2017) Os odontoideum: a comprehensive clinical and surgical review. Cureus 9(8):e1551. https://doi.org/10.7759/cureus.1551
Betsch MW, Blizzard SR, Shinseki MS, Yoo JU (2015) Prevalence of degenerative changes of the atlanto-axial joints. Spine J 15(2):275–280. https://doi.org/10.1016/j.spinee.2014.09.011
Wang S, Wang C, Yan M, Zhou H, Dang G (2013) Novel surgical classification and treatment strategy for atlantoaxial dislocations. Spine (Phila Pa 1976) 38(21):E1348–E1356. https://doi.org/10.1097/BRS.0b013e3182a1e5e4
Goel A (2019) Atlantoaxial instability: evolving understanding. Acta Neurochir Suppl 125:59–62. https://doi.org/10.1007/978-3-319-62515-7_9
Akobo S, Rizk E, Loukas M, Chapman JR, Oskouian RJ, Tubbs RS (2015) The odontoid process: a comprehensive review of its anatomy, embryology, and variations. Childs Nerv Syst 31(11):2025–2034. https://doi.org/10.1007/s00381-015-2866-4
Perdikakis E, Skoulikaris N (2014) The odontoid process: various configuration types in MR examinations. Eur Spine J 23(5):1077–1083. https://doi.org/10.1007/s00586-013-3135-4
Smoker WR (1994) Craniovertebral junction: normal anatomy, craniometry, and congenital anomalies. Radiographics 14(2):255–277. https://doi.org/10.1148/radiographics.14.2.8190952
Guille JT, Sherk HH (2002) Congenital osseous anomalies of the upper and lower cervical spine in children. J Bone Joint Surg Am 84(2):277–288. https://doi.org/10.2106/00004623-200202000-00017
Takeda E, Hashimoto T, Tayama M, Miyazaki M, Shirakawa E, Shiino Y, Saijo T, Ito M, Naito E, Huq AH, Et A (1991) Diagnosis of atlantoaxial subluxation in Morquio’s syndrome and spondyloepiphyseal dysplasia congenita. Acta Paediatr Jpn 33(5):633–638. https://doi.org/10.1111/j.1442-200x.1991.tb01877.x
Osti M, Philipp H, Meusburger B, Benedetto KP (2006) Os odontoideum with bipartite atlas and segmental instability: a case report. Eur Spine J 15(Suppl 5):564–567. https://doi.org/10.1007/s00586-005-0017-4
Funding
Supported by the Natural Science Basic Research Program of Shaanxi Province (2019JM-517), the Science and Technology Development Fund of Shaanxi Provincial People’s Hospital (2019YXQ-10) and the Key Research and Development projection of Shaanxi Province (2018ZDXM-SF-054).
Author information
Authors and Affiliations
Contributions
Ming Ling and Zhankui Jin designed the study. Yanhai Chang and Shizhang Liu screened the patients and diagnosed with KBD. Cuipei Hao and Zhi Yi reviewed and collected the patients’ medical data. Zhengming Sun and Zhehui Zhu searched relevant literature. Xueyuan Wu analyzed the data collected and wrote the manuscript. Ming Ling and Zhankui Jin approved the final version of the manuscript. The authors read and approved the final manuscript.
Corresponding author
Ethics declarations
Ethical approval
The study was approved by the Human and Ethics Committee for Medical Research of Shaanxi Provincial People’s Hospital. No organization sponsored the research.
Consent to participate
Informed consent was obtained from all the participants.
Conflict of interest
The authors declare no competing interests.
Additional information
Publisher's note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Rights and permissions
Springer Nature or its licensor (e.g. a society or other partner) holds exclusive rights to this article under a publishing agreement with the author(s) or other rightsholder(s); author self-archiving of the accepted manuscript version of this article is solely governed by the terms of such publishing agreement and applicable law.
About this article
Cite this article
Wu, X., Hao, C., Ling, M. et al. Prevalence and radiographic features of atlantoaxial dislocation in adult patients with Kashin–Beck disease. International Orthopaedics (SICOT) 47, 781–791 (2023). https://doi.org/10.1007/s00264-022-05616-6
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s00264-022-05616-6