The Spine Journal 9 (2009) e13–e17

Case Report

Long-term results of surgical treatment of dysphagia secondaryto cervical diffuse idiopathic skeletal hyperostosis

Julio Urrutia, MDa,*, Christopher M. Bono, MDb

aDepartment of Orthopaedic Surgery, Pontificia Universidad Catolica de Chile, Marcoleta 352, Santiago, Region Metropolitana, ChilebHarvard Medical School, Department of Orthopaedic Surgery, Brigham and Women’s Hospital, 75 Francis Street, Boston, MA 02115, USA

Received 23 December 2008; accepted 6 April 2009

Abstract BACKGROUND CONTEXT: Large, promine

FDA drug/device

Author disclosure

(royalties, Life Spine;

mor Danek; speaking

Stryker Spine; board

and/or materials, Arc

Spine; fellowship sup

* Corresponding a

E-mail address: ju

1529-9430/09/$ – see

doi:10.1016/j.spinee.2

nt osteophytes along the anterior aspect of the cervicalspine have been reported as a cause of dysphagia. Improvement of swallowing after surgical resectionhas been reported in a few case reports with short-term follow-up. The current report describes outcomesof a series of five patients with surgical treatment for this rare disorder, with a long-term follow-up.PURPOSE: To study the clinical and radiographic outcomes of a case series of patients surgicallytreated for dysphagia secondary to cervical diffuse idiopathic skeletal hyperostosis (DISH).STUDY DESIGN: Retrospective review of a case series.PATIENT SAMPLE: Five cases from a University Hospital.OUTCOME MEASURES: Clinical and imagenological follow-up.METHODS: The records of five patients with dysphagia who had undergone anterior surgical re-section of prominent osteophytes secondary to DISH were reviewed. Extrinsic esophageal compres-sion secondary to anterior cervical osteophytes was radiographically confirmed via preoperativebarium esophagogram swallowing study. All patients underwent anterior cervical osteophytes re-section without fusion. Postoperatively, patients were followed-up clinically and radiographicallywith routine lateral cervical radiographs.RESULTS: Preoperative esophagogram showed that the esophageal obstruction was present at onelevel in three cases and two levels in two cases. The C3–C4 level was involved in three cases, C4–C5 in three cases, and C5–C6 in one case. There were no postoperative complications, includingrecurrent laryngeal nerve palsy, wound infection, or hematomas. All patients had resolution of dy-phagia soon after surgery (within 2 weeks). Postoperative radiographs demonstrated complete re-moval of osteophytes. At final follow-up, ranging from 1 to 9 years (average 59.8 months,median 53 months), no patients reported recurrence of dysphagia. Final radiographic examinationdemonstrated minimal regrowth of the osteophytes.CONCLUSIONS: Although rarely indicated, surgical resection of anterior cervical osteophytesfrom DISH causing dyphagia produces good clinical and radiographical outcomes. After thoroughevaluation to rule out other intrinsic or extrinsic causes of swallowing difficulty, surgical treatmentof this uncommon condition might be considered. � 2009 Elsevier Inc. All rights reserved.

Keywords: Diffuse idiopathic skeletal hyperostosis (DISH); Cervical spondylotic dysphagia; Cervical osteophytes;

Surgical treatment

status: not applicable.

s: JU (consulting fees, Simpirica Spine); CMB

consulting fees, Depuy Spine and Medtronic Sofa-

and/or teaching arrangements, Depuy Spine and

of directors, North American Spine Society; staff

hus Orthopedics and Synthes Spine; grants, Stryker

port, Depuy Spine).

uthor. Tel.: (56) 2-2070101; fax: (56) 2-2067148.

rrutia@med.puc.cl (J. Urrutia)

front matter � 2009 Elsevier Inc. All rights reserved.

009.04.006

Introduction

Diffuse idiopathic skeletal hyperostosis (DISH), alsoknown as Forestier’s disease, is a noninflammatory enthesop-athy of unknown etiology. Affecting predominantly men, itresults in flowing, robust ossification of the anterior longitu-dinal ligament of the spine [1]. In contrast to ankylosingspondylitis, the disc space itself is usually spared, and, bydefinition, it affects four or more intervertebral levels [1–3].

Fig. 1. Esophagram showing extrinsecal compression secondary to ante-

rior cervical osteophytes (arrow).

e14 J. Urrutia and C.M. Bono / The Spine Journal 9 (2009) e13–e17

Notwithstanding postoperative complications, dyspha-gia, presenting as swallowing difficulty, is an uncommonsymptom intrinsically associated with a cervical spine dis-order. Despite its rarity, large anterior cervical osteophytescan be a primary cause of swallowing difficulty [4,5]. Os-teophytes can develop secondary to degenerative spondylo-sis or DISH, with the latter usually resulting in largerprominences [6,7].

Dyphagia secondary to anterior cervical osteophyteshave been frequently documented, primarily in isolatedcase reports [4,5,8–26]. Nonoperative management, includ-ing diet modification and medications, are the mainstay oftreatment [27,28]. However, in those cases in which nonop-erative modalities have failed and intrinsic esophageal dys-function has been ruled out, surgical resection of theosteophytes has been suggested as an effective, albeit lastresort, treatment [4,5,27,29–33].

Previous surgical reports have detailed good results ina limited number of cases [4,5,27,29–33], most them onshort follow-up. The authors present a case series withthe results of surgical excision of anterior cervical osteo-phytes for dyphagia in a homogenous group of patientswith DISH, with a long follow-up.

Material and methods

In a retrospective review of the authors’ operative re-cords at his institution between 1998 and 2008, five patientswere identified has having undergone surgical resection ofanterior cervical osteophytes for dysphagia with DISH. In-stitutional review board approval was obtained before con-ducting this study. The average age of the patients was 71years (range, 62–79) and all were men.

In each case, the patient had been previously seen by a gas-troenterologist and an otolaryngolist (Ears, Nose, and Throatspecialist) as part of a comprehensive diagnostic and thera-peutic evaluation of dysphagia. Importantly, other causesof dysphagia were ruled out, including intrinsic esophagealdysfunction. Patients underwent diagnostic endoscopy,which did not show evidence of any inner esophageal luminalabnormality, but did demonstrate extrinsic compression ineach case. In addition, a barium contrast esophagram wasmade, which confirmed esophageal compression secondaryto anterior cervical osteophytes (Fig. 1). Nonoperative man-agement was attempted in all patients, including diet modifi-cation, medication, and other modalities. Patients were thenreferred to the authors’ spine clinic for possible spine surgicalevaluation.

Cervical spine evaluation was performed, including a de-tailed history, physical examination, and anteroposteriorand lateral radiographs (Fig. 2). The diagnosis of DISHwas based on inspection of these radiographs, using the fol-lowing definition: flowing, nonmarginal bridging osteo-phytes involving four or more levels (Fig. 3). Otherdiagnoses, such as ankylosing spondylitis, inflammatory

arthritides, and degenerative spondylosis were ruled out us-ing these radiographs, among other tests (eg, blood tests,pelvic X-rays). Notably, disc spaces and the sacroiliacjoints were spared. Before presentation to the authors’spine clinic, patients had not been previously diagnosedwith DISH.

Preoperative counseling included an in-depth discussionof the potential risks and benefits of surgery with each pa-tient. Upon consenting to surgery, the preoperative esopha-gogram was used to determine which spinal levels werecausing extrinsic luminal compression.

Surgical technique

A standard Smith-Robinson anterior approach to thespine was performed through a longitudinal incision. Thislongitudinal approach allowed a longer exposure of the cer-vical spine, avoiding an excessive tension on an alreadycompressed esophagus. The esophagus was gently retractedmedially through blunt dissection to prevent any injury.With the anterior aspect of the spine exposed, a markerwas placed to identify the operative levels. To help betteridentify the deformed esophagus, a nasogastric tube was in-serted by the anesthesiologist so that it could be palpatedduring dissection.

Once the correct level(s) were identified, the longus collimuscles were subperiosteally elevated off the anterior spineand retracted laterally. Next, a midline trough was createdthrough the osteophytes using a high-speed burr. Carewas taken to differentiate between the relatively avascularosteophytic overgrowth and the more vascular vertebralbody. The annulus fibrosis was spared. The lateral aspectsof the osteophytes were removed using a Leksell rongeurin piecemeal fashion, while carefully protected the lateral

Fig. 2. Lateral preoperative cervical spine radiographs from three different patients showing large anterior cervical osteophytes.

e15J. Urrutia and C.M. Bono / The Spine Journal 9 (2009) e13–e17

soft-tissue structures, until flush with the midline trough.Bone bleeding was controlled with the use of bone wax. Af-ter final inspection of the esophagus, the wound was closedin layers over a surgical drain.

Postoperative management included mobilization as tol-erated on postoperative Day 1 and removal of the drain onpostoperative Day 1 as well. A liquid diet was initiated thesame day of surgery, and soft diet started on postoperativeDay 1. As symptoms resolved, the patients started a regulardiet. No cervical collar was used.

Follow-up evaluation

Patients were seen in the clinic after discharge from thehospital at 1 week, 1 month, and yearly after surgery.

Fig. 3. Typical diffuse idiopathic skeletal hyperostosis case showing flow-

ing, nonmarginal bridging osteophytes involving four or more levels.

Anteroposterior and lateral cervical radiographs were madeat each visit. A musculoskeletal radiologist evaluated all ra-diographs for the presence or absence of recurrentosteophytes.

Results

Follow-up ranged from 12 to 104 months (nearly 9years), with an average of 60 months and a median of 53months. Preoperative esophagogram demonstrated extrinsicluminal compression at one level in three of the patientsand two levels in two of the patients. The levels involvedwere C3–C4 in three cases, C4–C5 in three cases, andC5–C6 in one case.

There were no perioperative or postoperative complica-tions observed. Although in the hospital, patients tolerateda soft puree diet. Complete resolution of dysphagia was re-ported between 5 and 20 days after surgery, with an aver-age of 10 days, by which time a full, regular diet was welltolerated. This outcome was maintained at latest follow-up.

Immediate postoperative radiographs demonstratedcomplete removal of the osteophytes at the symptomaticlevels as measured by the true anterior vertebral body line(Fig. 4). Radiographs at latest follow-up demonstratedminimal regrowth of the osteophytes (Fig. 5). There wereno cases of new onset instability, spondylolisthesis, ordeformity.

Discussion

Anterior cervical osteophytes causing dysphagia havebeen frequently reported in the literature [4,6,9–17,34–39]. However, dysphagia from osteophytic overgrowthfrom DISH is less common [7,12,34,37,40–45]. In general,most patients can be treated nonoperatively [27–29].

Fig. 4. Immediate postoperative radiographs demonstrating complete re-

moval of the osteophytes at the symptomatic levels as measured by the true

anterior vertebral body line.

Fig. 5. Radiographs 104 months after surgery (longest follow-up) show-

ing minimal regrowth of the osteophytes.

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Patients who ultimately elect or require surgical inter-vention represent the most severe cases, though there arefew published series of outcomes [4,5,27,29–33]. Lauset al. [27] published a series of six patients with dysphagiasecondary to cervical osteophytes. The three most severecases underwent surgical treatment that resulted in rapid res-olution of symptoms, which was maintained at 1- to 2-yearfollow-up. Likewise, Goel et al. [29] reported three casesmanaged by surgical resection of cervical spondylotic osteo-phytes causing dysphagia, also documenting excellentresults at short-term follow-up.

Most reports of surgical treatment have used a standard,Smith-Robinson approach to the anterior cervical spine forosteophytectomy as used by the current authors. Using thistechnique, Humphreys et al. [6,30] reported good short-termresults in three patients in two separately published case re-ports. Likewise, Yee et al. [33] and Sobol and Rigual [46] pub-lished good long-term outcomes of two cases and one case,respectively, with the same approach. Of note, the transoral/transpharyngeal approach has also been used, albeit lessfrequently, to excise anterior cervical osteophytes [44]. Asthe relative indications and level of complexity of these twoapproaches vary considerably, a comparison between themhas thus far not been made.

In light of prior reports, the current authors present theoutcomes of a larger series of surgical resection of anteriorcervical osteophytes causing dysphagia. It carries the addi-tional distinction of being dedicated to a homogenousgroup of patients with DISH. Though previous reports sim-ilarly demonstrated reliable symptom resolution, they in-cluded only short-term follow-up. This left in questionwhether or not, and how quickly, DISH osteophytes can

reform and lead to a recurrence of symptoms. In contrastto these previous reports, the current series reported fol-low-up as long as 8.5 years. This suggests that symptom re-lief is well maintained over time.

Although the current report may represent the best avail-able evidence, drawing strong conclusions regarding thesafety and efficacy of surgical resection of osteophytes caus-ing cervical dysphagia is cautioned. Although the currentwork, as well as others, reports few or no adverse events,there are a number of complications that can potentially oc-cur and should be discussed with patients preoperatively. Aswith all retropharyngeal anterior approaches, there is a riskfor recurrent laryngeal and superior laryngeal nerve palsiesand operatively induced dysphagia, which may be difficultto differentiate from preoperative dysphagia. Although ex-ceedingly rare during elective anterior cervical surgery,the risk of iatrogenic esophageal tear may be higher thanusual because of the marked deformity and compressionby osteophytes.

Although not used in the current cases, there may bea role for administration of nonsteroidal anti-inflammatorymedication to prevent recurrence of osteophyte growth.Indomethacin has been recommended after total hipreplacement in patients with heterotopic ossification.Bisphosphonates had, at one time, been considered as pro-phylaxis after hip replacements in patients with DISH in-volving the spine, although the necessity of this practicehas since been refuted [47,48]. The use of such pharmaceu-ticals that have the potential to increase bleeding, whichmight cause a postoperative hematoma, should be carefullyconsidered. Because intervertebral fusion is not an integralpart of the described procedure, pseudarthrosis should notbe of clinical concern.

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