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Surgical Neurology 72 (2009) 266 – 271 www.surgicalneurology-online.com
Spine
Dysphagia due to anterior cervical hyperosteophytosis Mark E. Oppenlander, MD a , Daniel A. Orringer, MD b , Frank La Marca, MD b , John E. McGillicuddy, MD b , Stephen E. Sullivan, MD b , William F. Chandler, MD b , Paul Park, MD b,⁎ a
University of Michigan Medical School, Ann Arbor, MI, 3552 Taubman Center, Box 0338, 1500 East Medical Center Drive, Ann Arbor, MI 48109, USA b Department of Neurosurgery, University of Michigan Health System, Ann Arbor, MI 48109-0338, USA Received 10 July 2008; accepted 26 August 2008
Abstract
Background: Anterior cervical hyperosteophytosis describes the excessive formation of osteophytes along the ventral spine. Dysphagia due to ACH is considered an uncommon entity described mainly in case reports. Symptomatic ACH has been attributed to multiple etiologies including DISH, trauma, postlaminectomy syndromes, and cervical spondylosis. We report one of the largest series of patients with ACH-induced dysphagia requiring surgery. Methods: After IRB approval, a retrospective chart review was completed. From 2001 to 2006, 9 patients presented with dysphagia due to ACH requiring surgical treatment. Results: Eight patients were male, and the mean age was 65.1 years. Cervical spine x-rays and CT clearly demonstrated ACH in each case. Esophagram or a video fluoroscopic swallowing study was used to verify that dysphagia was caused by osteophytic overgrowth in all instances but one. In 2 patients, a focal osteophyte had formed adjacent to a previously fused segment. Of the remaining 7 patients, osteophytic formation was attributed to cervical spondylosis in 2 patients and DISH in 5 patients. All patients underwent osteophytectomy without spinal fusion. Average follow-up was 9.8 months. Although all 9 patients experienced resolution of dysphagia, improvement was delayed in 2 patients. Conclusions: Diffuse idiopathic skeletal hyperostosis and spondylosis are the most common etiologies accounting for ACH-induced dysphagia. Adjacent segment disease may also be a potential cause of symptomatic ACH and has not been previously reported. Regardless of etiology, surgical resection is highly successful if conservative measures fail. © 2009 Elsevier Inc. All rights reserved.
Keywords:
Anterior cervical hyperosteophytosis; Cervical osteophyte; Dysphagia; Osteophytectomy; Surgery
1. Introduction Anterior cervical hyperosteophytosis describes the excessive formation of osteophytes along the ventral spine. Dysphagia due to ACH is considered an uncommon entity
Abbreviations: ACH, anterior cervical hyperosteophytosis; CT, computed tomography; DISH, diffuse idiopathic skeletal hyperostosis; IRB, institutional review board. ⁎ Corresponding author. Tel.: +1 734 615 2627; fax: +1 734 936 9294. E-mail address:
[email protected] (P. Park). 0090-3019/$ – see front matter © 2009 Elsevier Inc. All rights reserved. doi:10.1016/j.surneu.2008.08.081
and has been described mainly in case reports. The osteophytic overgrowth causing dysphagia has been attributed to multiple etiologies including DISH, trauma, postlaminectomy syndromes, and cervical spondylosis [6,23,31,37-39,48]. Although various mechanisms have been suggested, the predominant cause of dysphagia is thought to be mechanical compression of the esophagus by overgrown osteophytes. Symptomatic ACH has been treated by osteophytectomy, which is the surgical resection of the osteophytes. This study represents our institutional experience with dysphagia caused by ACH and underscores the multiple etiologies of ACH. Most notably, focal ACH attributed to
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adjacent segment disease is described, which previously has not been reported. In addition, diagnostic evaluation, surgical treatment, and outcomes are reviewed. 2. Patients and methods University of Michigan Health Systems IRB approval was obtained before completing a retrospective chart review. From 2001 to 2006, 9 patients presented with dysphagia as a primary symptom requiring surgical treatment. Significant ACH was noted in all cases. The dysphagia was attributed to ACH and verified by either a video fluoroscopic swallowing study or esophogram in all cases but one. For each patient, demographic data, radiographic studies, surgical treatment, and outcomes were analyzed. 2.1. Surgical technique All patients in this study underwent osteophytectomy without fusion. An anterolateral extrapharyngeal approach, typically used for an anterior cervical discectomy and fusion, was used in each case. A tissue plane was developed medial to the sternocleidomastoid muscle to allow lateral retraction of the carotid sheath and contralateral retraction of the esophagus and trachea to expose the ventral spine. Using fluoroscopy to determine the extent of resection, we used rongeurs and a highspeed drill to remove the symptomatic osteophytes and reestablish a more normal contour to the spine (Fig. 1).
3. Results Of the 9 patients evaluated, 8 were men and the mean age was 65.1 years (range, 40-77 years) (Table 1). In addition to dysphagia as the presenting symptom, 1 patient also had neck pain and another complained of respiratory stridor. Two patients developed symptomatic ACH at the segment adjacent to a prior fusion. Of these, 1 patient had a history of a C3-7 fusion and presented 5 years later with dysphagia due to a focal C2-3 osteophyte. The second patient had previously undergone a C2-5 fusion and presented approximately 21 years later with significant ACH in the segments caudal to the fusion. The adjacent C5-6 segment appeared most problematic. Symptomatic ACH was attributed to DISH in 5 patients and spondylosis in the remaining 2 patients. As part of the diagnostic evaluation, all patients had anterior-posterior and lateral plain x-rays of the cervical spine. Six patients had CT. Seven patients underwent a video fluoroscopic swallowing study and 2 patients underwent an esophagram to confirm obstruction by the osteophytic overgrowth. In all cases but one, osteophytic obstruction was verified. In one patient, the esophagram did not demonstrate an obstruction. However, his x-rays and CT revealed extensive ACH, which prompted a recommendation for surgery. The distribution of symptomatic ACH varied. In the subgroup of patients with DISH, osteophytic growth diffusely involved the spine. However, symptomatic ACH
Fig. 1. A: Preoperative lateral x-ray demonstrating significant ACH causing dysphagia. B: Postoperative lateral x-ray after surgical resection of the symptomatic osteophytes.
involved the C2-5 segments in 2 patients and the C2-6, C3-6, and C5-7 segments in the remaining 3 patients, respectively. For the 2 patients with spondylosis, symptomatic ACH affected the C3-6 and C4-7 segments. In the patients with ACH attributed to adjacent segment disease, the C2-3 level in one patient and primarily the C5-6 level in the other were symptomatic. Overall, the most commonly involved level was C4-5.
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Table 1 Patients diagnosed with ACH-induced dysphagia and treated by osteophytectomy Patient
Age (y)
Sex
Presenting symptom(s)
Etiology
Affected spinal levels
Follow-up (m)
Outcome
1 2 3 4 5 6 7 8 9
58 40 72 77 59 78 65 72 65
F M M M M M M M M
Dysphagia Dysphagia, neck pain Dysphagia Dysphagia Dysphagia Dysphagia Dysphagia, stridor Dysphagia Dysphagia
ASD (previous C3-7 fusion) ASD (previous C2-5 fusion) DISH2 Spondylosis DISH DISH DISH Spondylosis DISH
C2-C3 C5-T2 a C5-C7 C3-C6 C3-C6 C2-C5 C2-C5 C4-7 C2-6
20 6 9 10 3 1 5 29 5
Full resolution Full resolution Delayed resolution Full resolution Full resolution Full resolution Delayed resolution Full resolution Full resolution
ASD indicates adjacent segment disease; F, female; M, male. a 5-6 predominant osteophyte, modest osteophytic formation involving C6-T2.
The mean length of hospital stay after surgery was 3 days (range, 1-9 days). One patient developed atrial fibrillation, delaying his discharge until postoperative day 5. Another patient had persistent dysphagia postoperatively, necessitating the placement of a gastrostomy tube for feeding and delaying his discharge until day 9. This patient had undergone a C2-5 osteophytectomy, and his persistent dysphagia was attributed to edema. The mean follow-up was 9.8 months (range, 129 months). All patients experienced significant improvement in their dysphagia. Seven patients noted immediate improvement in symptoms and by their 1-month follow-up visit were able to consume a normal diet. Of the remaining 2 patients, one was the previously noted patient requiring a gastrostomy tube. This patient had persistent dysphagia until the 2-month follow-up visit when improvement was noted. At the 4-month follow-up visit, the patient was able to tolerate a normal diet. The last patient had undergone a C35 osteophytectomy and had persistent dysphagia at the time of discharge on postoperative day 2. However, by the 1month follow-up visit, improvement was noted, and the patient subsequently was able to tolerate a near normal diet with the exclusion of certain solid foods. 4. Discussion Dysphagia due to ACH is considered relatively uncommon. This study describes one of the largest series to date of patients with ACH-induced dysphagia requiring surgery and highlights the multiple etiologies underlying the development of symptomatic osteophytosis. Mainly noted in case reports, DISH is cited as one of the most common causes of ACH-induced dysphagia requiring surgical management [5-7,9-14,17,18,20,23,25,27,32-35,42,47]. Patients with DISH develop a flowing ossification of the anterior longitudinal ligament that results in bridging osteophytes. Also known as Forestier disease, DISH is an idiopathic condition characterized by spinal and peripheral enthesopathy, most notably ossification of the anterior longitudinal ligament. The condition was first described as its own entity by Forestier and Rotes-Querol in 1950 as senile ankylosing hyperostosis of the spine [16]. Subsequently, Resnick et al
[44] emphasized the extraspinal manifestations of the disease and more appropriately referred to the condition as DISH. Radiographically, 3 major criteria for the diagnosis of DISH have been proposed: (1) the presence of flowing ossification along the anterolateral aspect of at least 4 contiguous vertebral bodies; (2) relative preservation of intervertebral disk heights with minimal disk degeneration; (3) absence of apophyseal joint ankylosis and sacroiliac joint sclerosis to rule out an inflammatory etiology [43]. Diffuse idiopathic skeletal hyperostosis is a relatively common condition that occurs with aging and has been found in 12% to 28% of autopsy specimens [3,43]. On average, patients with DISH are in the sixth decade of life, and there is a male predominance [3,28,43,49]. Consistent with these demographic characteristics, the 5 patients with DISH in our study had an average age of 67.8 years (range, 59-72 years) and all were men. Although any region of the spine can be affected, one relatively large clinical study noted cervical spine involvement in 53 (78%) of 68 patients with DISH [43]. The incidence of dysphagia in one study of 30 DISH patients was 17% [49]. Resnick et al found a 28% incidence of dysphagia in a study of 21 DISH patients but noted that only 10% required surgical resection. Spondylosis is also a common finding in the aging cervical spine. Although most osteophytic growth from spondylosis is asymptomatic, the development of significant osteophytes causing dysphagia has been reported [1,2,8,21,22,29]. Typically more focal than DISH with regard to the extent of osteophyte formation, loss of disk space height is also a distinguishing factor. Focal osteophytic formation after trauma and postlaminectomy instability have also been reported as causes of dysphagia [31,39]. One etiology not previously reported is the development of symptomatic ACH due to adjacent segment disease. Adjacent segment disease describes the development of degenerative changes at the mobile segment next to a spinal fusion. Biomechanical studies of human cadaveric cervical spines have shown increased strain at the adjacent levels after 1-, 2-, and 3-level fixation, suggesting a mechanism for accelerated degeneration of the remaining motion segments after fusion [41]. Clinically, Robertson et al [45] noted an increased incidence of adjacent segment disease in patients
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Fig. 2. Preoperative x-ray of a 40-year-old patient with history of a previous C2-5 fusion, who presented with severe dysphagia attributed primarily to the C5-6 osteophyte. After surgical resection, the patient was able to tolerate a normal diet.
who underwent cervical fusion compared to those who maintained motion with artificial disk placement. Another study of 112 patients demonstrated symptomatic degenerative changes of the segment adjacent to a cervical fusion in 19% of patients [26]. Presumably, accelerated degenerative changes can result in anterior osteophyte formation, which is the etiology proposed for 2 of the patients in our study. In support of an accelerated degenerative process, the 2 patients with ACH-induced dysphagia attributed to adjacent segment disease were the youngest patients in our study at 40 and 58 years of age (Fig. 2). In addition, the symptomatic osteophyte was focal, involving the segment immediately adjacent to the fusion in each of the 2 cases. A variety of mechanisms can account for ACH-induced dysphagia [4,7,32,40,46]: (1) mechanical obstruction by an enlarged osteophyte compressing or deviating the esophageal lumen; (2) growth of a relatively small osteophyte at sites where the esophagus is anatomically anchored (level of the cricoid cartilage and diaphragm); (3) inflammation of the soft tissue due to repetitive motion across the osteophyte promoting esophageal obstruction; (4) pain and muscle spasm from an enlarged osteophyte causing esophageal narrowing; (5) osteophyte-induced restriction in mobility of the epiglottis as well as limitation in movement of the larynx; (6) indentation of pharynx wall by an osteophyte resulting in retention of food in piriform sinus. In addition to complete history and physical examination, diagnostic evaluation should include cervical spine x-rays. Although x-rays often will clearly demonstrate ACH, CT may be useful to better delineate the bony anatomy, particularly in the patient with an unusual body habitus. To confirm diagnosis
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of ACH-induced dysphagia, an esophagram or video fluoroscopic swallowing study should be performed. Treatment of ACH-induced dysphagia, particularly in patients with milder symptoms, should begin with conservative management consisting of diet modification, swallow therapy, nonsteroidal anti-inflammatory medication, steroids, muscle relaxants, and/or sedation [4,15,24,30,40]. In patients where the mechanism of dysphagia is from osteophyteinduced inflammation, pain, or muscle spasms, conservative measures are most likely to succeed. However, in the patient with refractory dysphagia or in one who presents with severe dysphagia, surgical osteophytectomy is the most effective option. In a literature review of surgical resection of anterior cervical osteophytes causing dysphagia, 28 (93%) of 30 patients noted symptomatic improvement after surgery [15]. Although 2 patients had delayed improvement, all 9 patients in our study noted significant relief of dysphagia after surgical resection. This delayed improvement was attributed to postoperative edema from retraction of the esophagus. However, McCafferty et al [36] has suggested that delayed improvement may reflect esophageal inflammation and/or fibrosis that persists after removal of the symptomatic osteophyte. Although the complication rate appears low, specific risks associated with the anterolateral extrapharyngeal approach for osteophyte resection include vocal cord paresis/ paralysis, Horner syndrome, and esophageal or tracheal perforation with fistula and/or abscess formation caused by thinning of the walls from chronic osteophytic compression [4,19,30]. The recurrence rate for ACH-induced dysphagia is unclear and is likely dependent on the underlying etiology. Although none of the patients in our study has had a recurrence, symptomatic osteophytic regrowth has been noted to occur in one study involving 2 patients with DISH at 5.4 and 7 years, respectively, after the initial surgical resection [15]. 5. Conclusions Diffuse idiopathic skeletal hyperostosis and spondylitic disease are the most commonly noted etiologies for ACHinduced dysphagia. Adjacent segment disease also appears to be a potential cause of symptomatic osteophytes. Regardless of etiology, however, osteophytectomy consisting of surgical resection of the enlarged osteophytes is usually highly successful if conservative measures fail. References [1] Bauer F. Dysphagia due to cervical spondylosis. J Laryngol Otol 1953; 67(10):615-30. [2] Blumberg P, Prapote C, Viscomi G. Cervical osteophytes producing dysphagia. Ear Nose Throat J 1977;56:396-9. [3] Boachie-Adjei O, Bullough PG. Incidence of ankylosing hyperostosis of the spine (Forestier's disease) at autopsy. Spine 1987;12(8):739-43. [4] Brandenberg G, Leibrock LG. Dysphagia and dysphonia secondary to anterior cervical osteophytes. Neurosurgery 1986;18(1):90-3. [5] Burkus JK. Esophageal obstruction secondary to diffuse idiopathic skeletal hyperostosis. Orthopedics 1988;11(5):717-20.
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Commentary This article is an excellent overview of a relatively rare but clinically pertinent condition, which most neurosurgeons will encounter sooner or later. That this condition can develop adjacent to long cervical fusions has not been previously reported and is a consideration for patient and surgeon when such fusions are indicated. The various mechanisms by which the condition becomes symptomatic speak to the complex functions within the anterior cervical compartment.
