- Email: [email protected]
Surgical management of Eagle syndrome: A 17-year experience with open and transoral robotic styloidectomy
Am J Otolaryngol xxx (xxxx) xxxx
Contents lists available at ScienceDirect
Am J Otolaryngol journal homepage: www.elsevier.com/locate/amjoto
Surgical management of Eagle syndrome: A 17-year experience with open and ransoral robotic styloidectomy ⁎
Thomas H. Fitzpatrick IVa, , Benjamin D. Lovinb, Marcus J. Magisterc, Joshua D. Waltonenc, J. Dale Brownec, Christopher A. Sullivanc a
Wake Forest University School of Medicine, 475 Vine St, Winston-Salem, NC 27101, USA Baylor College of Medicine, Bobby R. Alford Department of Otolaryngology-Head and Neck Surgery, One Baylor Plaza, Houston, TX 77030, USA c Wake Forest University Baptist Medical Center, Department of Otolaryngology-Head and Neck Surgery, Medical Center Boulevard, Winston-Salem, NC 27157, USA b
A B S T R A C T
Eagle Syndrome (ES) is a rare disorder that can present with symptoms ranging from globus sensation to otalgia that is attributed to an elongated styloid process and/ or calcified stylohyoid ligament. No standardized treatment algorithm exists, and although various surgical approaches have been described, data on the use of transoral robotic surgery (TORS) in this population is limited. To investigate the utility of TORS in the treatment of ES, a retrospective review in 19 ES patients was carried out at a single academic, tertiary medical center between 2000 and 2017. Nineteen patients underwent twenty-one styloid resections: 6 performed via TORS and 15 via transcervical approach. Across all patients, 90% reported some degree of lasting improvement in symptoms while 55% reported significant improvement. When TORS was compared to transcervical resection, there was no difference in the subjective rate of “meaningful” (83 vs. 57%) versus rate of “non-meaningful” symptom improvement (17 vs. 43%) (p = 0.35). There was a trend towards less estimated blood loss (EBL), operative time, and post-operative length of stay (LOS) with TORS versus transcervical cases (9.2 mL vs. 30.0 mL, 98 vs. 156 min, and 0.7 vs. 1.2 days); however, these did not reach statistical significance (p = .11, 0.13, and 0.42, respectively). Three patients experienced complications associated with an open approach, as compared to none with TORS. In select patients, TORS styloidectomy is a reasonable surgical alternative to traditional transoral and transcervical techniques as it provides similar symptom improvement, and reduced length of stay, blood loss, and operative time.
1. Introduction Eagle Syndrome (ES) is a rare condition caused by symptomatic elongation of the styloid process and/or calcification of the stylohyoid ligament. These anatomic variations lead to local irritation and inflammation of the glossopharyngeal nerve [1]. ES was first described by Dr. Watt Eagle in 1937 [2] and is characterized by a constellation of cervicofacial symptoms that can include throat pain, dysphagia, dysphonia, globus sensation, and otalgia [3]. Elongation of the styloid process (> 30 mm [4]) occurs in ~4% of the general population, but only ~4–10% of those patients develop symptoms [5]. As originally described, there are two subtypes of ES—classic and vascular. The classic subtype is considered to be caused by prior surgery, whereas the vascular or carotid artery syndrome subtype is caused by impingement of the styloid process on the carotid nerve plexus and is unrelated to prior surgery [6,7]. Eagle originally proposed that prior tonsillectomy might be the overwhelming etiology in the classic subtype [8], but no study to date has been able support this hypothesis [9]. ES is a diagnosis of exclusion and is reached by a thorough history, physical exam, and imaging. Physical exam maneuvers include
palpation of the tonsillar fossa for an elongated styloid process and reproducible symptoms with head turning [3]. Furthermore, symptom resolution upon injection of a local anesthetic into the tonsillar fossa can be diagnostic [4]. Clinical suspicion should then be confirmed by evidence of an elongated styloid process on computed tomography (CT). Of note, medial angulation, not length of the styloid process, has recently been shown to correlate most closely with symptom severity [10]. Symptom management of ES begins with conservative medical treatment, such as anti-inflammatory medications, anti-convulsants, anti-psychotics, or other analgesics [11]; however, definitive and longlasting management has long been accomplished surgically [12] by either a transoral or transcervical styloidectomy. Transoral resection results in no external scar and can be accomplished in a shorter time than an open transcervical approach [13]. The transoral approach, however, can be limited by poor exposure and lighting thus increasing risk of injury to critical neurovascular structures [14]. As such, the use of transoral robotic surgery (TORS) for styloidectomy has begun to appear in the literature due to technical advantages of superior endoscopic lighting and instrument maneuverability [11]. Despite these
⁎
Corresponding author. E-mail addresses: tfi[email protected] (T.H. Fitzpatrick), [email protected] (B.D. Lovin), [email protected] (M.J. Magister), [email protected] (J.D. Waltonen), [email protected] (J.D. Browne), [email protected] (C.A. Sullivan). https://doi.org/10.1016/j.amjoto.2019.102324 Received 4 October 2019 0196-0709/ © 2019 Elsevier Inc. All rights reserved.
Please cite this article as: Thomas H. Fitzpatrick, et al., Am J Otolaryngol, https://doi.org/10.1016/j.amjoto.2019.102324
Am J Otolaryngol xxx (xxxx) xxxx
T.H. Fitzpatrick, et al.
inherent advantages, there remain only a few case reports and a small case series that report the use of the robot for styloidectomy [11,15,16]. To date, the present study represents the largest cohort of ES patients to undergo TORS styloidectomy and the first to critically assess its utility alongside the “gold standard,” open approach. The objective of this study was to evaluate and compare surgical treatment approaches for ES at a single tertiary care hospital over a 17year period.
Table 1 Patient characteristics.
N Age at surgery (years) Female Caucasian African American Hispanic Asian Prior tonsillectomy Length of styloid process (mm) Width of styloid process (mm) Medial angulation (degrees) Mucosal depth (mm)
2. Material and methods The described study is a retrospective chart review. Clinical information was collected from the electronic medical record (EMR) of patients who had undergone styloidectomy. The setting for this study was Wake Forest University Baptist Medical Center, a tertiary referral, academic medical center. Patients were identified with the diagnosis of ES between 2000 and 2017 by querying the EMR system for the following International Classification of Disease (ICD), 9th Revision and ICD, 10th Revision, codes: 352.1, 733.99, G52.1, and M89.8X8. From this query, 25 patients were found to have a diagnosis of classic subtype ES. Of these 25 patients, 19 underwent either transcervical or TORS styloidectomy. Two patients had bilateral resections via the transcervical approach, for a total of 15 transcervical styloidectomies. In this study, bilateral cases were counted once in determining demographic factors and presenting symptoms, but separately for all other calculations. Three different otolaryngologists performed all styloidectomies in the patient population, two of which performed all of the TORS cases (CS, JW). The robotic system used in all TORS cases was the da Vinci SI (Intuitive Surgical, Sunnyvale, CA). The EMR for each patient identified was reviewed to extract data including patient demographics, characteristics and severity of symptoms, radiographic data, treatment, estimated blood loss (EBL), length of stay in hospital (LOS), operative duration, and complications. Based on symptom change as reported by patients in post-operative visits, patients were labeled as significant (near complete resolution), moderate, mild, temporary improvement, no change, or worsened symptoms. In comparing the clinical effectiveness of each surgical technique, these categories were condensed into two groups: significant and moderate improvement was designated as “meaningful” improvement, and all other results were categorized as “non-meaningful” improvement. To measure medial angulation, a vertical line was passed through the midline of the odontoid process and another line was passed along the styloid process as seen on coronal CT. The angle between the two lines was recorded as depicted in Fig. 2, with a smaller angle denoting greater medial deviation. Styloid process width was measured on axial CT scans at the skull base, and pharyngeal mucosa depth was measured from the distal tip of the styloid. Length was measured using sagittal scans. Patients were stratified by operative technique. The choice of approach was based upon the practice pattern and clinical skill set of each surgeon while considering general patient suitability for a robotic approach such as body habitus, orofacial anatomy, location of the carotid artery, and willingness to undergo a transoral robotic approach. Data was presented as either mean ± standard deviation for continuous variables or count (percent) for categorical variables. Categorical data between techniques was evaluated using the Fisher exact test. Differences in continuous data between techniques were calculated using the student's t-test, with the Satterthwaite adjustment for unequal variances between groups in comparing EBL and operative duration.
Overall
TORS
Transcervical
19 47.5 ± 15.2 15 (79%) 14 (74%) 3 (16%) 1 (5%) 1 (5%) 4 (21%) 37.5 ± 10.1
6 49.3 ± 10.4 5 (83%) 5 (83%) 1 (17%) 0 (0%) 0 (0%) 0 (0%) 41.3 ± 9.4
13 46.7 ± 17.3 10 (77%) 9 (69%) 2 (15%) 1 (8%) 1 (8%) 4 (30%) 35.4 ± 10.2
0.25 0.25
5.4 ± 1.7
5.9 ± 2.0
5.2 ± 1.6
0.5
23.1 ± 4.1
19.8 ± 2.4
25.1 ± 3.6
0.01*
7.2 ± 4.7
9.5 ± 5.6
5.8 ± 3.7
0.18
0 (0%) 5 (83%) 1 (17%)
1 (8%) 9 (69%) 3 (23%)
1
Pre-operative symptom severity Mild 1 (5%) Moderate 14 (74%) Severe 4 (21%)
p-Value
0.84 1 1
patients underwent a bilateral procedure for a total of 21 styloid process resections. Median follow-up post-intervention for all patients was 63 days. Of the 21 styloidectomies, 6 were performed via TORS and 15 were done transcervically. No statistical differences were found in demographics or pre-operative symptom severity between groups (Table 1). Across all patients, Caucasian race and female gender were most common, with average age being 48 ± 15 years (Table 1). The mean length of the styloid process for all patients was 37.5 ± 10.1 mm, width 5.4 ± 1.7 mm, and medial angulation 23.1 ± 4.1° (Table 1). There was no difference in styloid process length or width between groups (p = .25, 0.5), but medial angulation was significantly different between the TORS and transcervical approaches (19.8° vs. 25.1°, p = .01) (Table 1). The depth of the pharyngeal mucosa to the distal end of the styloid was 9.5 mm in the TORS group vs. 5.8 mm for transcervical, but this result did not reach statistical significance (p = .18). The most common presenting symptoms were throat pain, otalgia, and neck pain (Table 2). Of all 21 resections, 90% reported some degree of lasting symptom improvement with 55% reporting significant improvement. Of the TORS patients, 100% reported some degree of lasting symptomatic improvement and 66% reported significant improvement (Fig. 1). When comparing techniques, there was no difference in patients reporting meaningful versus non-meaningful improvement (p = 0.35) (Table 3). There was a trend towards less EBL, less operative time, and shorter post-operative LOS in the TORS group compared to transcervical group (9.2 vs. 30.0 mL, 98 vs. 156 min, and 0.7 vs. 1.2 days, respectively); these did not, however, reach statistical significance (p = 0.11, 0.13, and 0.42, respectively) (Table 4). In total, only three patients (14%) experienced complications, which included cellulitis, trigeminal neuralgia, cranial nerve (CN) XII weakness, and CN XI weakness. There were no complications in the TORS group.
Table 2 Primary presenting symptoms.
Throat pain Otalgia Neck pain Globus sensation
3. Results Twenty-five patients with ES were identified. Of those, 19 underwent styloidectomy and were included in the present study. Two 2
Overall
TORS
Transcervical
p-value
10 (56%) 8 (44%) 7 (39%) 6 (33%)
3 3 3 1
7 5 4 5
0.765
(50%) (50%) (50%) (17%)
(58%) (42%) (33%) (42%)
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Table 4 Operative details.
Cases EBL (mL) Operative duration (min) Hospital LOS (days) Complication rate
Table 3 Post-operative symptom change.
Meaningful improvement Non-meaningful improvement
TORS
Transcervical
p-value
13 (65%) 7 (35%)
5 (83%) 1 (17%)
8 (57%) 6 (43%)
0.35
TORS
Transcervical
p-value
21 21.1 ± 26.5 132 ± 73 1.0 ± 1.3 3 (14%)
6 9.2 ± 3.8 98 ± 22 0.7 ± 0.5 0 (0%)
15 30.0 ± 31.6 156 ± 90 1.2 ± 1.6 3 (20%)
0.11 0.13 0.42 0.53
approaches due to the small population and the introduction of TORS at a later point. With further study, we expect a cutoff to exist, where a deeper styloid would be less amenable to the TORS approach. Additionally, only four patients in this study (21%) had previously undergone tonsillectomy, which supports existing data that Eagle's hypothesis of tonsillectomy as the most likely inciting factor in the classic subtype is unlikely; idiopathic development appears to be the most common etiology [9]. The most common symptoms at presentation were otalgia, throat pain, and neck pain. This data closely mirrors that by Hardin et al. in their retrospective review of 21 patients treated with either transcervical or non-TORS, transoral styloidectomy [20]. They similarly found that almost half of patients reported ear and throat pain; however, their rate of neck pain was much higher—specifically, 80% versus 37% in the present study [20]. These results are important as a large percentage of referred patients who are ultimately diagnosed with ES may come from dentistry after TMJ has been ruled out as a cause for jaw or facial pain [21]. The surgical approaches in this study addressed ES symptoms equally well. This confirms a prior, similarly powered study evaluating the efficacy of surgical intervention in ES patients [20]. Upon stratifying by technique, TORS exhibited an excellent rate of symptom improvement—100% demonstrated some degree of improvement and 66% demonstrated significant improvement. Similar improvement was recently reported in a similar yet smaller case series by Kim et al. which included 4 cases of TORS styloidectomy [15]. The present data serves to consistently demonstrate the benefit of this approach though should be interpreted with caution since medial angulation of the styloid and not surgical technique may be the actual reason for success using the robotic approach. Finally, this is the first study to directly compare outcomes of TORS and transcervical styloidectomy. In our cohort, there was a slight trend towards an increased rate of “meaningful” improvement in the TORS group; this trend did not reach statistical significance (p = .35). This lack of significant difference is not surprising as the etiologic determinant of symptoms in ES is similarly resected in each technique. TORS, however, often allows styloidectomy to be performed with less dissection and disruption of surrounding tissues thereby decreasing risk of inadvertent surgical complications.
Fig. 1. Representative calculation of medial angulation.
Overall
Overall
4. Discussion ES is a rare disorder that presents with vague head and neck symptoms attributed to an elongated styloid process and/or calcified stylohyoid ligament [1]. Medical management has traditionally given way to surgical styloidectomy as a lasting means of symptom resolution [17]. Transcervical or transoral styloidectomy have been the traditional approaches, but TORS is emerging in select patients due to potential shortcomings associated with other approaches [18,19]. The technical advantages offered by TORS include endoscopic visualization, reliable lighting, magnification, and improvement in fine motor control while operating in the vicinity of critical neurovascular structures [11]. There were no differences observed in the demographics of each surgical group, apart from medial angulation of the elongated styloid process. This result was expected as TORS would be the intuitive surgical approach to a more medially deflected styloid as it would be closer to the pharyngeal mucosa. It is likely that the recorded pharyngeal mucosa depth in this study was not significantly different between
Fig. 2. Distribution of symptom improvement. 3
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option in select patients that provides symptomatic improvement with little blood loss, decreased operative time, shorter LOS and lower complication rates when compared to other approaches.
Complications in the present study included cellulitis and neuropathies (2 of which were of cranial nerves). These complications were seen in the transcervical group with none observed in the TORS group. Though the TORS cohort contained fewer patients, this outcome is expected since TORS provides improved visibility and identification of vital neurovascular structures, as well as omitting the morbidity associated with open surgery. Similarly, Kim et al. found no intraoperative or post-operative complications in their TORS cohort [15]. In assessing potential differences in operative/hospital associated factors, the use of TORS demonstrated a trend towards less EBL, operative time (including set-up), and LOS, though statistical significance was not reached. The trend towards shorter LOS can potentially be attributed to earlier initiation of oral intake in TORS patients, as Kim et al. found that all 4 of their patients restarted an oral diet on postoperative day 1. Though the present study did not specifically investigate time to initiation of PO diet, all patients were discharged on an oral diet, without the need for enteral feeding. Despite the potential benefit of using TORS in these cases, it is important to consider that access to this surgical approach is limited not only by surgical training, but also by the cost burden of the robotic system. As such, further research on the cost-effectiveness of this approach is warranted. Potential limitation associated with this study includes its retrospective nature. As such, the study population is limited, especially given the low incidence of ES. As our hospital system converted to an EMR in the middle of the study, some data that was accessible for the more recent cases was unable to be determined for earlier cases. Additionally, some patients underwent multiple procedures at the time of their styloidectomy, which directly affected EBL and operative duration. Finally, given the retrospective nature of this study, selection bias between techniques is likely and the approach for each patient was based on each surgeon's preference, TORS eligibility criteria, and other patient specific factors (ie. BMI). Despite these limitations, this study is valuable as it represents the largest collections of ES patients to undergo styloidectomy via TORS, as well as the only study to compare TORS and transcervical styloidectomy. It also serves to validate similar findings reported in the literature. These results support generalizability of the TORS approach in selected patients with sufficiently medially deflected styloid processes at centers with robot availability and appropriately experienced robotic surgeons.
Acknowledgements No source of funding, no conflicts of interest to disclose. References [1] Eagle WW. Elongated styloid process: symptoms and treatment. JAMA Otolaryngology–Head & Neck Surgery 1958;67(2):172–6. [2] Eagle WW. Elongated styloid processes: report of two cases. JAMA Otolaryngology–Head & Neck Surgery 1937;25(5):584–7. [3] Galletta K, et al. Eagle syndrome: a wide spectrum of clinical and neuroradiological findings from cervico-facial pain to cerebral ischemia. J Craniofac Surg 2019;30(5):e424–8. [4] Badhey A, et al. Eagle syndrome: a comprehensive review. Clin Neurol Neurosurg 2017;159:34–8. [5] Papadiochos I, et al. Treatment of Eagle syndrome with transcervical approach secondary to a failed intraoral attempt: surgical technique and literature review. J Stomatol Oral Maxillofac Surg 2017;118(6):353–8. [6] Eagle WW. Symptomatic elongated styloid process report of two cases of Styloid process–carotid artery syndrome with operation. JAMA Otolaryngology–Head & Neck Surgery 1949;49(5):490–503. [7] Mendelsohn AH, Berke GS, Chhetri DK. Heterogeneity in the clinical presentation of Eagle’s syndrome. Otolaryngol Head Neck Surg 2006;134(3):389–93. [8] Fusco DJ, Asteraki S, Spetzler RF. Eagle’s syndrome: embryology, anatomy, and clinical management. Acta Neurochir 2012;154(7):1119–26. [9] FRITZ M. Elongated styloid process: a cause of obscure throat symptoms. JAMA Otolaryngology–Head & Neck Surgery 1940;31(6):911–8. [10] Okur A, et al. Is there a relationship between symptoms of patients and tomographic characteristics of styloid process? Surg Radiol Anat 2014;36(7):627–32. [11] Montevecchi F, et al. Transoral robotic surgery (TORS) for bilateral Eagle syndrome. ORL J Otorhinolaryngol Relat Spec 2019;81(1):36–40. [12] Mortellaro C, et al. Eagle’s syndrome: importance of a corrected diagnosis and adequate surgical treatment. J Craniofac Surg 2002;13(6):755–8. [13] Buono U, et al. Surgical approach to the stylohyoid process in Eagle’s syndrome. J Oral Maxillofac Surg 2005;63(5):714–6. [14] Muderris T, et al. Surgical management of elongated styloid process: intraoral or transcervical? Eur Arch Otorhinolaryngol 2014;271(6):1709–13. [15] Kim DH, et al. Transoral robotic surgery in Eagle's syndrome: our experience on four patients. Acta Otorhinolaryngol Ital 2017;37(6):454–7. [16] Kadakia S, et al. Tonsillectomy sparing transoral robot assisted styloidectomy. Am J Otolaryngol 2018;39(2):238–41. [17] Fini G, et al. The long styloid process syndrome or Eagle’s syndrome. J Craniomaxillofac Surg 2000;28(2):123–7. [18] Ankur Kirankumar Walli VPT, Parelkar K, Nagle S, Kulsange KL. Intraoral styloidectomy in Eagle’s syndrome-a risky and infrequently performed approach. J Clin Diagn Res 2018;12(1):MD01–2. [19] Jalisi S, Jamal BT, Grillone GA. Surgical management of long-standing Eagle’s syndrome. Ann Maxillofac Surg 2017;7(2):232–6. [20] Hardin FM, Xiao R, Burkey BB. Surgical management of patients with Eagle syndrome. Am J Otolaryngol 2018;39(5):481–4. [21] Keur JJ, et al. The clinical significance of the elongated styloid process. Oral Surg Oral Med Oral Pathol 1986;61(4):399–404.
5. Conclusions Diagnosis of ES can prove challenging not only for the Otolaryngologist, but also for primary care providers given its rarity and range of presentation; however, when accurately diagnosed, operative intervention can lead to consistent and significant symptomatic improvement. TORS should be considered a safe, minimally invasive
4
Contents lists available at ScienceDirect
Am J Otolaryngol journal homepage: www.elsevier.com/locate/amjoto
Surgical management of Eagle syndrome: A 17-year experience with open and ransoral robotic styloidectomy ⁎
Thomas H. Fitzpatrick IVa, , Benjamin D. Lovinb, Marcus J. Magisterc, Joshua D. Waltonenc, J. Dale Brownec, Christopher A. Sullivanc a
Wake Forest University School of Medicine, 475 Vine St, Winston-Salem, NC 27101, USA Baylor College of Medicine, Bobby R. Alford Department of Otolaryngology-Head and Neck Surgery, One Baylor Plaza, Houston, TX 77030, USA c Wake Forest University Baptist Medical Center, Department of Otolaryngology-Head and Neck Surgery, Medical Center Boulevard, Winston-Salem, NC 27157, USA b
A B S T R A C T
Eagle Syndrome (ES) is a rare disorder that can present with symptoms ranging from globus sensation to otalgia that is attributed to an elongated styloid process and/ or calcified stylohyoid ligament. No standardized treatment algorithm exists, and although various surgical approaches have been described, data on the use of transoral robotic surgery (TORS) in this population is limited. To investigate the utility of TORS in the treatment of ES, a retrospective review in 19 ES patients was carried out at a single academic, tertiary medical center between 2000 and 2017. Nineteen patients underwent twenty-one styloid resections: 6 performed via TORS and 15 via transcervical approach. Across all patients, 90% reported some degree of lasting improvement in symptoms while 55% reported significant improvement. When TORS was compared to transcervical resection, there was no difference in the subjective rate of “meaningful” (83 vs. 57%) versus rate of “non-meaningful” symptom improvement (17 vs. 43%) (p = 0.35). There was a trend towards less estimated blood loss (EBL), operative time, and post-operative length of stay (LOS) with TORS versus transcervical cases (9.2 mL vs. 30.0 mL, 98 vs. 156 min, and 0.7 vs. 1.2 days); however, these did not reach statistical significance (p = .11, 0.13, and 0.42, respectively). Three patients experienced complications associated with an open approach, as compared to none with TORS. In select patients, TORS styloidectomy is a reasonable surgical alternative to traditional transoral and transcervical techniques as it provides similar symptom improvement, and reduced length of stay, blood loss, and operative time.
1. Introduction Eagle Syndrome (ES) is a rare condition caused by symptomatic elongation of the styloid process and/or calcification of the stylohyoid ligament. These anatomic variations lead to local irritation and inflammation of the glossopharyngeal nerve [1]. ES was first described by Dr. Watt Eagle in 1937 [2] and is characterized by a constellation of cervicofacial symptoms that can include throat pain, dysphagia, dysphonia, globus sensation, and otalgia [3]. Elongation of the styloid process (> 30 mm [4]) occurs in ~4% of the general population, but only ~4–10% of those patients develop symptoms [5]. As originally described, there are two subtypes of ES—classic and vascular. The classic subtype is considered to be caused by prior surgery, whereas the vascular or carotid artery syndrome subtype is caused by impingement of the styloid process on the carotid nerve plexus and is unrelated to prior surgery [6,7]. Eagle originally proposed that prior tonsillectomy might be the overwhelming etiology in the classic subtype [8], but no study to date has been able support this hypothesis [9]. ES is a diagnosis of exclusion and is reached by a thorough history, physical exam, and imaging. Physical exam maneuvers include
palpation of the tonsillar fossa for an elongated styloid process and reproducible symptoms with head turning [3]. Furthermore, symptom resolution upon injection of a local anesthetic into the tonsillar fossa can be diagnostic [4]. Clinical suspicion should then be confirmed by evidence of an elongated styloid process on computed tomography (CT). Of note, medial angulation, not length of the styloid process, has recently been shown to correlate most closely with symptom severity [10]. Symptom management of ES begins with conservative medical treatment, such as anti-inflammatory medications, anti-convulsants, anti-psychotics, or other analgesics [11]; however, definitive and longlasting management has long been accomplished surgically [12] by either a transoral or transcervical styloidectomy. Transoral resection results in no external scar and can be accomplished in a shorter time than an open transcervical approach [13]. The transoral approach, however, can be limited by poor exposure and lighting thus increasing risk of injury to critical neurovascular structures [14]. As such, the use of transoral robotic surgery (TORS) for styloidectomy has begun to appear in the literature due to technical advantages of superior endoscopic lighting and instrument maneuverability [11]. Despite these
⁎
Corresponding author. E-mail addresses: tfi[email protected] (T.H. Fitzpatrick), [email protected] (B.D. Lovin), [email protected] (M.J. Magister), [email protected] (J.D. Waltonen), [email protected] (J.D. Browne), [email protected] (C.A. Sullivan). https://doi.org/10.1016/j.amjoto.2019.102324 Received 4 October 2019 0196-0709/ © 2019 Elsevier Inc. All rights reserved.
Please cite this article as: Thomas H. Fitzpatrick, et al., Am J Otolaryngol, https://doi.org/10.1016/j.amjoto.2019.102324
Am J Otolaryngol xxx (xxxx) xxxx
T.H. Fitzpatrick, et al.
inherent advantages, there remain only a few case reports and a small case series that report the use of the robot for styloidectomy [11,15,16]. To date, the present study represents the largest cohort of ES patients to undergo TORS styloidectomy and the first to critically assess its utility alongside the “gold standard,” open approach. The objective of this study was to evaluate and compare surgical treatment approaches for ES at a single tertiary care hospital over a 17year period.
Table 1 Patient characteristics.
N Age at surgery (years) Female Caucasian African American Hispanic Asian Prior tonsillectomy Length of styloid process (mm) Width of styloid process (mm) Medial angulation (degrees) Mucosal depth (mm)
2. Material and methods The described study is a retrospective chart review. Clinical information was collected from the electronic medical record (EMR) of patients who had undergone styloidectomy. The setting for this study was Wake Forest University Baptist Medical Center, a tertiary referral, academic medical center. Patients were identified with the diagnosis of ES between 2000 and 2017 by querying the EMR system for the following International Classification of Disease (ICD), 9th Revision and ICD, 10th Revision, codes: 352.1, 733.99, G52.1, and M89.8X8. From this query, 25 patients were found to have a diagnosis of classic subtype ES. Of these 25 patients, 19 underwent either transcervical or TORS styloidectomy. Two patients had bilateral resections via the transcervical approach, for a total of 15 transcervical styloidectomies. In this study, bilateral cases were counted once in determining demographic factors and presenting symptoms, but separately for all other calculations. Three different otolaryngologists performed all styloidectomies in the patient population, two of which performed all of the TORS cases (CS, JW). The robotic system used in all TORS cases was the da Vinci SI (Intuitive Surgical, Sunnyvale, CA). The EMR for each patient identified was reviewed to extract data including patient demographics, characteristics and severity of symptoms, radiographic data, treatment, estimated blood loss (EBL), length of stay in hospital (LOS), operative duration, and complications. Based on symptom change as reported by patients in post-operative visits, patients were labeled as significant (near complete resolution), moderate, mild, temporary improvement, no change, or worsened symptoms. In comparing the clinical effectiveness of each surgical technique, these categories were condensed into two groups: significant and moderate improvement was designated as “meaningful” improvement, and all other results were categorized as “non-meaningful” improvement. To measure medial angulation, a vertical line was passed through the midline of the odontoid process and another line was passed along the styloid process as seen on coronal CT. The angle between the two lines was recorded as depicted in Fig. 2, with a smaller angle denoting greater medial deviation. Styloid process width was measured on axial CT scans at the skull base, and pharyngeal mucosa depth was measured from the distal tip of the styloid. Length was measured using sagittal scans. Patients were stratified by operative technique. The choice of approach was based upon the practice pattern and clinical skill set of each surgeon while considering general patient suitability for a robotic approach such as body habitus, orofacial anatomy, location of the carotid artery, and willingness to undergo a transoral robotic approach. Data was presented as either mean ± standard deviation for continuous variables or count (percent) for categorical variables. Categorical data between techniques was evaluated using the Fisher exact test. Differences in continuous data between techniques were calculated using the student's t-test, with the Satterthwaite adjustment for unequal variances between groups in comparing EBL and operative duration.
Overall
TORS
Transcervical
19 47.5 ± 15.2 15 (79%) 14 (74%) 3 (16%) 1 (5%) 1 (5%) 4 (21%) 37.5 ± 10.1
6 49.3 ± 10.4 5 (83%) 5 (83%) 1 (17%) 0 (0%) 0 (0%) 0 (0%) 41.3 ± 9.4
13 46.7 ± 17.3 10 (77%) 9 (69%) 2 (15%) 1 (8%) 1 (8%) 4 (30%) 35.4 ± 10.2
0.25 0.25
5.4 ± 1.7
5.9 ± 2.0
5.2 ± 1.6
0.5
23.1 ± 4.1
19.8 ± 2.4
25.1 ± 3.6
0.01*
7.2 ± 4.7
9.5 ± 5.6
5.8 ± 3.7
0.18
0 (0%) 5 (83%) 1 (17%)
1 (8%) 9 (69%) 3 (23%)
1
Pre-operative symptom severity Mild 1 (5%) Moderate 14 (74%) Severe 4 (21%)
p-Value
0.84 1 1
patients underwent a bilateral procedure for a total of 21 styloid process resections. Median follow-up post-intervention for all patients was 63 days. Of the 21 styloidectomies, 6 were performed via TORS and 15 were done transcervically. No statistical differences were found in demographics or pre-operative symptom severity between groups (Table 1). Across all patients, Caucasian race and female gender were most common, with average age being 48 ± 15 years (Table 1). The mean length of the styloid process for all patients was 37.5 ± 10.1 mm, width 5.4 ± 1.7 mm, and medial angulation 23.1 ± 4.1° (Table 1). There was no difference in styloid process length or width between groups (p = .25, 0.5), but medial angulation was significantly different between the TORS and transcervical approaches (19.8° vs. 25.1°, p = .01) (Table 1). The depth of the pharyngeal mucosa to the distal end of the styloid was 9.5 mm in the TORS group vs. 5.8 mm for transcervical, but this result did not reach statistical significance (p = .18). The most common presenting symptoms were throat pain, otalgia, and neck pain (Table 2). Of all 21 resections, 90% reported some degree of lasting symptom improvement with 55% reporting significant improvement. Of the TORS patients, 100% reported some degree of lasting symptomatic improvement and 66% reported significant improvement (Fig. 1). When comparing techniques, there was no difference in patients reporting meaningful versus non-meaningful improvement (p = 0.35) (Table 3). There was a trend towards less EBL, less operative time, and shorter post-operative LOS in the TORS group compared to transcervical group (9.2 vs. 30.0 mL, 98 vs. 156 min, and 0.7 vs. 1.2 days, respectively); these did not, however, reach statistical significance (p = 0.11, 0.13, and 0.42, respectively) (Table 4). In total, only three patients (14%) experienced complications, which included cellulitis, trigeminal neuralgia, cranial nerve (CN) XII weakness, and CN XI weakness. There were no complications in the TORS group.
Table 2 Primary presenting symptoms.
Throat pain Otalgia Neck pain Globus sensation
3. Results Twenty-five patients with ES were identified. Of those, 19 underwent styloidectomy and were included in the present study. Two 2
Overall
TORS
Transcervical
p-value
10 (56%) 8 (44%) 7 (39%) 6 (33%)
3 3 3 1
7 5 4 5
0.765
(50%) (50%) (50%) (17%)
(58%) (42%) (33%) (42%)
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Table 4 Operative details.
Cases EBL (mL) Operative duration (min) Hospital LOS (days) Complication rate
Table 3 Post-operative symptom change.
Meaningful improvement Non-meaningful improvement
TORS
Transcervical
p-value
13 (65%) 7 (35%)
5 (83%) 1 (17%)
8 (57%) 6 (43%)
0.35
TORS
Transcervical
p-value
21 21.1 ± 26.5 132 ± 73 1.0 ± 1.3 3 (14%)
6 9.2 ± 3.8 98 ± 22 0.7 ± 0.5 0 (0%)
15 30.0 ± 31.6 156 ± 90 1.2 ± 1.6 3 (20%)
0.11 0.13 0.42 0.53
approaches due to the small population and the introduction of TORS at a later point. With further study, we expect a cutoff to exist, where a deeper styloid would be less amenable to the TORS approach. Additionally, only four patients in this study (21%) had previously undergone tonsillectomy, which supports existing data that Eagle's hypothesis of tonsillectomy as the most likely inciting factor in the classic subtype is unlikely; idiopathic development appears to be the most common etiology [9]. The most common symptoms at presentation were otalgia, throat pain, and neck pain. This data closely mirrors that by Hardin et al. in their retrospective review of 21 patients treated with either transcervical or non-TORS, transoral styloidectomy [20]. They similarly found that almost half of patients reported ear and throat pain; however, their rate of neck pain was much higher—specifically, 80% versus 37% in the present study [20]. These results are important as a large percentage of referred patients who are ultimately diagnosed with ES may come from dentistry after TMJ has been ruled out as a cause for jaw or facial pain [21]. The surgical approaches in this study addressed ES symptoms equally well. This confirms a prior, similarly powered study evaluating the efficacy of surgical intervention in ES patients [20]. Upon stratifying by technique, TORS exhibited an excellent rate of symptom improvement—100% demonstrated some degree of improvement and 66% demonstrated significant improvement. Similar improvement was recently reported in a similar yet smaller case series by Kim et al. which included 4 cases of TORS styloidectomy [15]. The present data serves to consistently demonstrate the benefit of this approach though should be interpreted with caution since medial angulation of the styloid and not surgical technique may be the actual reason for success using the robotic approach. Finally, this is the first study to directly compare outcomes of TORS and transcervical styloidectomy. In our cohort, there was a slight trend towards an increased rate of “meaningful” improvement in the TORS group; this trend did not reach statistical significance (p = .35). This lack of significant difference is not surprising as the etiologic determinant of symptoms in ES is similarly resected in each technique. TORS, however, often allows styloidectomy to be performed with less dissection and disruption of surrounding tissues thereby decreasing risk of inadvertent surgical complications.
Fig. 1. Representative calculation of medial angulation.
Overall
Overall
4. Discussion ES is a rare disorder that presents with vague head and neck symptoms attributed to an elongated styloid process and/or calcified stylohyoid ligament [1]. Medical management has traditionally given way to surgical styloidectomy as a lasting means of symptom resolution [17]. Transcervical or transoral styloidectomy have been the traditional approaches, but TORS is emerging in select patients due to potential shortcomings associated with other approaches [18,19]. The technical advantages offered by TORS include endoscopic visualization, reliable lighting, magnification, and improvement in fine motor control while operating in the vicinity of critical neurovascular structures [11]. There were no differences observed in the demographics of each surgical group, apart from medial angulation of the elongated styloid process. This result was expected as TORS would be the intuitive surgical approach to a more medially deflected styloid as it would be closer to the pharyngeal mucosa. It is likely that the recorded pharyngeal mucosa depth in this study was not significantly different between
Fig. 2. Distribution of symptom improvement. 3
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option in select patients that provides symptomatic improvement with little blood loss, decreased operative time, shorter LOS and lower complication rates when compared to other approaches.
Complications in the present study included cellulitis and neuropathies (2 of which were of cranial nerves). These complications were seen in the transcervical group with none observed in the TORS group. Though the TORS cohort contained fewer patients, this outcome is expected since TORS provides improved visibility and identification of vital neurovascular structures, as well as omitting the morbidity associated with open surgery. Similarly, Kim et al. found no intraoperative or post-operative complications in their TORS cohort [15]. In assessing potential differences in operative/hospital associated factors, the use of TORS demonstrated a trend towards less EBL, operative time (including set-up), and LOS, though statistical significance was not reached. The trend towards shorter LOS can potentially be attributed to earlier initiation of oral intake in TORS patients, as Kim et al. found that all 4 of their patients restarted an oral diet on postoperative day 1. Though the present study did not specifically investigate time to initiation of PO diet, all patients were discharged on an oral diet, without the need for enteral feeding. Despite the potential benefit of using TORS in these cases, it is important to consider that access to this surgical approach is limited not only by surgical training, but also by the cost burden of the robotic system. As such, further research on the cost-effectiveness of this approach is warranted. Potential limitation associated with this study includes its retrospective nature. As such, the study population is limited, especially given the low incidence of ES. As our hospital system converted to an EMR in the middle of the study, some data that was accessible for the more recent cases was unable to be determined for earlier cases. Additionally, some patients underwent multiple procedures at the time of their styloidectomy, which directly affected EBL and operative duration. Finally, given the retrospective nature of this study, selection bias between techniques is likely and the approach for each patient was based on each surgeon's preference, TORS eligibility criteria, and other patient specific factors (ie. BMI). Despite these limitations, this study is valuable as it represents the largest collections of ES patients to undergo styloidectomy via TORS, as well as the only study to compare TORS and transcervical styloidectomy. It also serves to validate similar findings reported in the literature. These results support generalizability of the TORS approach in selected patients with sufficiently medially deflected styloid processes at centers with robot availability and appropriately experienced robotic surgeons.
Acknowledgements No source of funding, no conflicts of interest to disclose. References [1] Eagle WW. Elongated styloid process: symptoms and treatment. JAMA Otolaryngology–Head & Neck Surgery 1958;67(2):172–6. [2] Eagle WW. Elongated styloid processes: report of two cases. JAMA Otolaryngology–Head & Neck Surgery 1937;25(5):584–7. [3] Galletta K, et al. Eagle syndrome: a wide spectrum of clinical and neuroradiological findings from cervico-facial pain to cerebral ischemia. J Craniofac Surg 2019;30(5):e424–8. [4] Badhey A, et al. Eagle syndrome: a comprehensive review. Clin Neurol Neurosurg 2017;159:34–8. [5] Papadiochos I, et al. Treatment of Eagle syndrome with transcervical approach secondary to a failed intraoral attempt: surgical technique and literature review. J Stomatol Oral Maxillofac Surg 2017;118(6):353–8. [6] Eagle WW. Symptomatic elongated styloid process report of two cases of Styloid process–carotid artery syndrome with operation. JAMA Otolaryngology–Head & Neck Surgery 1949;49(5):490–503. [7] Mendelsohn AH, Berke GS, Chhetri DK. Heterogeneity in the clinical presentation of Eagle’s syndrome. Otolaryngol Head Neck Surg 2006;134(3):389–93. [8] Fusco DJ, Asteraki S, Spetzler RF. Eagle’s syndrome: embryology, anatomy, and clinical management. Acta Neurochir 2012;154(7):1119–26. [9] FRITZ M. Elongated styloid process: a cause of obscure throat symptoms. JAMA Otolaryngology–Head & Neck Surgery 1940;31(6):911–8. [10] Okur A, et al. Is there a relationship between symptoms of patients and tomographic characteristics of styloid process? Surg Radiol Anat 2014;36(7):627–32. [11] Montevecchi F, et al. Transoral robotic surgery (TORS) for bilateral Eagle syndrome. ORL J Otorhinolaryngol Relat Spec 2019;81(1):36–40. [12] Mortellaro C, et al. Eagle’s syndrome: importance of a corrected diagnosis and adequate surgical treatment. J Craniofac Surg 2002;13(6):755–8. [13] Buono U, et al. Surgical approach to the stylohyoid process in Eagle’s syndrome. J Oral Maxillofac Surg 2005;63(5):714–6. [14] Muderris T, et al. Surgical management of elongated styloid process: intraoral or transcervical? Eur Arch Otorhinolaryngol 2014;271(6):1709–13. [15] Kim DH, et al. Transoral robotic surgery in Eagle's syndrome: our experience on four patients. Acta Otorhinolaryngol Ital 2017;37(6):454–7. [16] Kadakia S, et al. Tonsillectomy sparing transoral robot assisted styloidectomy. Am J Otolaryngol 2018;39(2):238–41. [17] Fini G, et al. The long styloid process syndrome or Eagle’s syndrome. J Craniomaxillofac Surg 2000;28(2):123–7. [18] Ankur Kirankumar Walli VPT, Parelkar K, Nagle S, Kulsange KL. Intraoral styloidectomy in Eagle’s syndrome-a risky and infrequently performed approach. J Clin Diagn Res 2018;12(1):MD01–2. [19] Jalisi S, Jamal BT, Grillone GA. Surgical management of long-standing Eagle’s syndrome. Ann Maxillofac Surg 2017;7(2):232–6. [20] Hardin FM, Xiao R, Burkey BB. Surgical management of patients with Eagle syndrome. Am J Otolaryngol 2018;39(5):481–4. [21] Keur JJ, et al. The clinical significance of the elongated styloid process. Oral Surg Oral Med Oral Pathol 1986;61(4):399–404.
5. Conclusions Diagnosis of ES can prove challenging not only for the Otolaryngologist, but also for primary care providers given its rarity and range of presentation; however, when accurately diagnosed, operative intervention can lead to consistent and significant symptomatic improvement. TORS should be considered a safe, minimally invasive
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