Research on effectiveness of occipito-odontoid angle in predicting dysphagia after occipitocervical fusion in patients with C<sub>2, 3</sub> Klippel-Feil syndrome_Chinese Journal of Reparative and Reconstructive Surgery

Authors：

ZOU Qiang , Wang Linnan ,  YANG Xi , CHEN Taiyong , HU Bowen , LIU Limin , SONG Yueming

Department of Orthopedics, Orthopedic Research Institute, West China Hospital, Sichuan University, Chengdu Sichuan, 610041, P. R. China;

Corresponding author：

YANG Xi, Email: formosa88@163.com

Keywords：

Occipitocervical fusion; C_{2, 3} Klippel-Feil syndrome; dysphagia; occipitocervical angle

DOI：

10.7507/1002-1892.202110004

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Objective To introduce a new occipitocervical angle parameter, occipito-odontoid angle (O-Da), for predicting dysphagia after occipitocervical fusion (OCF) in patients with C_{2, 3} Klippel-Feil syndrome (KFS) and analyze its effectiveness. Methods A total of 119 patients met selective criteria between April 2010 and November 2019 were retrospectively included as the study subjects. There were 56 males and 63 females. The age ranged from 14 to 76 years, with a median age of 51 years. There were 44 cases of basilar invagination and 75 cases of atlantoaxial subluxation. Forty patients were combined with C_{2, 3} KFS. Seven patients underwent anterior decompression combined with posterior OCF and 112 patients underwent posterior OCF. The fixed segments were O-C₂ in 36 cases, O-C₃ in 51 cases, O-C₄ in 25 cases, and O-C₅ in 7 cases. All patients were followed up 21-136 months, with a median time of 79 months. The lateral cervical X-ray films before operation and at last follow-up were used to measure the occipital to C₂ angle (O-C₂a), the occipital and external acoustic meatus to axis angle (O-EAa), the occipital protuberance to axial angle (Oc-Axa), the O-Da, and the narrowest oropharyngeal airway space (nPAS). The differences of the above parameters between the last follow-up and the preoperative values were calculated (represented as dO-C₂a, dO-EAa, dOc-Axa, dO-Da, and dnPAS). Patients were divided into two groups according to whether they suffered dysphagia after operation, and the differences in clinical data and radiographic parameters were compared between the two groups. The correlation between occipitocervical angle parameters and nPAS in 40 patients with C_{2, 3} KFS was analyzed respectively. In addition, sensitivity and specificity analyses were used to assess the effectiveness of dO-Da≤−5° for the prediction of postoperative dysphagia. Results Thirty-one patients (26.1%) suffered dysphagia after OCF (dysphagia group), including 10 patients with C_{2, 3} KFS; no dysphagia occurred in 88 patients (non-dysphagia group). There was no significant difference in age, follow-up time, fixed segment, proportion of patients with rheumatoid arthritis, proportion of patients with atlantoaxial subluxation, and proportion of patients with C_{2, 3} KFS between the two groups (P>0.05). The proportion of female patients was significantly higher in dysphagia group than in non-dysphagia group (χ²=7.600, P=0.006). The difference in preoperative O-C₂a between the two groups was significant (t=2.528, P=0.014). No significant differences were observed in preoperative O-EAa, Oc-Axa, O-Da, and nPAS (P>0.05). There was no significant difference in dO-C₂a, dO-EAa, dOc-Axa, dO-Da, and dnPAS between the two groups (P>0.05). The dO-C₂a, dO-EAa, dOc-Axa, and dO-Da were positively correlated with dnPAS in 40 patients with C_{2, 3} KFS (r=0.604, P<0.001; r=0.649, P<0.001; r=0.615, P<0.001; r=0.672, P<0.001). Taking dO-Da≤−5° as the standard, the sensitivity and specificity of dO-Da to predict postoperative dysphagia in patients with C_{2, 3} KFS were 80.0% (8/10) and 93.3% (28/30), respectively. Conclusion The dO-Da is a reliable indicator for predicting dysphagia after OCF in patients with C_{2, 3} KFS.

Citation： ZOU Qiang, Wang Linnan, YANG Xi, CHEN Taiyong, HU Bowen, LIU Limin, SONG Yueming. Research on effectiveness of occipito-odontoid angle in predicting dysphagia after occipitocervical fusion in patients with C_{2, 3} Klippel-Feil syndrome. Chinese Journal of Reparative and Reconstructive Surgery, 2022, 36(6): 739-744. doi: 10.7507/1002-1892.202110004 Copy

1.	Garrido BJ, Sasso RC. Occipitocervical fusion. Orthop Clin North Am, 2012, 43(1): 1-9.
2.	Lavano A, Guzzi G, Stroscio CA, et al. Occipitocervical fusion. Acta Neurochir Suppl, 2019, 125: 243-245.
3.	Hong J, Lim S. Dysphagia after occipitocervical fusion. N Engl J Med, 2017, 376(22): e46. doi: 10.1056/NEJMicm1610752.
4.	Izeki M, Neo M, Ito H, et al. Reduction of atlantoaxial subluxation causes airway stenosis. Spine (Phila Pa 1976), 2013, 38(9): E513-E520.
5.	Miyata M, Neo M, Fujibayashi S, et al. O-C₂ angle as a predictor of dyspnea and/or dysphagia after occipitocervical fusion. Spine (Phila Pa 1976), 2009, 34(2): 184-188.
6.	Yoshida M, Neo M, Fujibayashi S, et al. Upper-airway obstruction after short posterior occipitocervical fusion in a flexed position. Spine (Phila Pa 1976), 2007, 32(8): E267-E270.
7.	Morizane K, Takemoto M, Neo M, et al. Occipital and external acoustic meatus to axis angle as a predictor of the oropharyngeal space in healthy volunteers: a novel parameter for craniocervical junction alignment. Spine J, 2018, 18(5): 811-817.
8.	Ota M, Neo M, Aoyama T, et al. Impact of the O-C₂ angle on the oropharyngeal space in normal patients. Spine (Phila Pa 1976), 2011, 36(11): E720-E726.
9.	Wang LN, Hu BW, Song YM, et al. Predictive abilities of O-C₂a and O-EAa for the development of postoperative dysphagia in patients undergoing occipitocervical fusion. Spine J, 2020, 20(5): 745-753.
10.	Izeki M, Neo M, Takemoto M, et al. The O-C₂ angle established at occipito-cervical fusion dictates the patient’s destiny in terms of postoperative dyspnea and/or dysphagia. Eur Spine J, 2014, 23(2): 328-336.
11.	Meng Y, Wu T, Liu Z, et al. The impact of the difference in O-C₂ angle in the development of dysphagia after occipitocervical fusion: a simulation study in normal volunteers combined with a case-control study. Spine J, 2018, 18(8): 1388-1397.
12.	Chen T, Yang X, Kong W, et al. Impact of the occiput and external acoustic meatus to axis angle on dysphagia in patients suffering from anterior atlantoaxial subluxation after occipitocervical fusion. Spine J, 2019, 19(8): 1362-1368.
13.	Patel K, Evans H, Sommaruga S, et al. Characteristics and management of pain in patients with Klippel-Feil syndrome: analysis of a global patient-reported registry. J Neurosurg Spine, 2020, 32(4): 578-583.
14.	Corsello G, Carcione A, Castro L, et al. Zerviko-Okulo-Akustikus-Syndrom (Wildervanck-Syndrom): eine klinische Variante des Klippel-Feil-Syndroms? Klinische Padiatrie, 1990, (3): 176-179.
15.	Juberg RC, Gershanik JJ. Cervical vertebral fusion (Klippel-Feil) syndrome with consanguineous parents. J Med Genet, 1976, 13(3): 246-249.
16.	Gruber J, Saleh A, Bakhsh W, et al. The prevalence of Klippel-Feil syndrome: A computed tomography—Based analysis of 2, 917 patients. Spine Deformity, 2018, 6(4): 448-453.
17.	Nagashima S, Nagae M, Arai Y, et al. A new method of measuring the occipitocervical angle that could be applied as an intraoperative indicator during occipitocervical fusion. Clin Spine Surg, 2017, 30(7): E981-E987.
18.	Bazaz R, Lee MJ, Yoo JU. Incidence of dysphagia after anterior cervical spine surgery: a prospective study. Spine (Phila Pa 1976), 2002, 27(22): 2453-2458.

1. Garrido BJ, Sasso RC. Occipitocervical fusion. Orthop Clin North Am, 2012, 43(1): 1-9.
2. Lavano A, Guzzi G, Stroscio CA, et al. Occipitocervical fusion. Acta Neurochir Suppl, 2019, 125: 243-245.
3. Hong J, Lim S. Dysphagia after occipitocervical fusion. N Engl J Med, 2017, 376(22): e46. doi: 10.1056/NEJMicm1610752.
4. Izeki M, Neo M, Ito H, et al. Reduction of atlantoaxial subluxation causes airway stenosis. Spine (Phila Pa 1976), 2013, 38(9): E513-E520.
5. Miyata M, Neo M, Fujibayashi S, et al. O-C₂ angle as a predictor of dyspnea and/or dysphagia after occipitocervical fusion. Spine (Phila Pa 1976), 2009, 34(2): 184-188.
6. Yoshida M, Neo M, Fujibayashi S, et al. Upper-airway obstruction after short posterior occipitocervical fusion in a flexed position. Spine (Phila Pa 1976), 2007, 32(8): E267-E270.
7. Morizane K, Takemoto M, Neo M, et al. Occipital and external acoustic meatus to axis angle as a predictor of the oropharyngeal space in healthy volunteers: a novel parameter for craniocervical junction alignment. Spine J, 2018, 18(5): 811-817.
8. Ota M, Neo M, Aoyama T, et al. Impact of the O-C₂ angle on the oropharyngeal space in normal patients. Spine (Phila Pa 1976), 2011, 36(11): E720-E726.
9. Wang LN, Hu BW, Song YM, et al. Predictive abilities of O-C₂a and O-EAa for the development of postoperative dysphagia in patients undergoing occipitocervical fusion. Spine J, 2020, 20(5): 745-753.
10. Izeki M, Neo M, Takemoto M, et al. The O-C₂ angle established at occipito-cervical fusion dictates the patient’s destiny in terms of postoperative dyspnea and/or dysphagia. Eur Spine J, 2014, 23(2): 328-336.
11. Meng Y, Wu T, Liu Z, et al. The impact of the difference in O-C₂ angle in the development of dysphagia after occipitocervical fusion: a simulation study in normal volunteers combined with a case-control study. Spine J, 2018, 18(8): 1388-1397.
12. Chen T, Yang X, Kong W, et al. Impact of the occiput and external acoustic meatus to axis angle on dysphagia in patients suffering from anterior atlantoaxial subluxation after occipitocervical fusion. Spine J, 2019, 19(8): 1362-1368.
13. Patel K, Evans H, Sommaruga S, et al. Characteristics and management of pain in patients with Klippel-Feil syndrome: analysis of a global patient-reported registry. J Neurosurg Spine, 2020, 32(4): 578-583.
14. Corsello G, Carcione A, Castro L, et al. Zerviko-Okulo-Akustikus-Syndrom (Wildervanck-Syndrom): eine klinische Variante des Klippel-Feil-Syndroms? Klinische Padiatrie, 1990, (3): 176-179.
15. Juberg RC, Gershanik JJ. Cervical vertebral fusion (Klippel-Feil) syndrome with consanguineous parents. J Med Genet, 1976, 13(3): 246-249.
16. Gruber J, Saleh A, Bakhsh W, et al. The prevalence of Klippel-Feil syndrome: A computed tomography—Based analysis of 2, 917 patients. Spine Deformity, 2018, 6(4): 448-453.
17. Nagashima S, Nagae M, Arai Y, et al. A new method of measuring the occipitocervical angle that could be applied as an intraoperative indicator during occipitocervical fusion. Clin Spine Surg, 2017, 30(7): E981-E987.
18. Bazaz R, Lee MJ, Yoo JU. Incidence of dysphagia after anterior cervical spine surgery: a prospective study. Spine (Phila Pa 1976), 2002, 27(22): 2453-2458.

Chinese Journal of Reparative and Reconstructive Surgery

Research on effectiveness of occipito-odontoid angle in predicting dysphagia after occipitocervical fusion in patients with C_{2, 3} Klippel-Feil syndrome

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Research on effectiveness of occipito-odontoid angle in predicting dysphagia after occipitocervical fusion in patients with C2, 3 Klippel-Feil syndrome

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Research on effectiveness of occipito-odontoid angle in predicting dysphagia after occipitocervical fusion in patients with C_{2, 3} Klippel-Feil syndrome