Surgical management of dysphagia due to diffuse idiopathic skeletal hyperostosis: the role of barium swallow fluoroscopy—a case report

Introduction

Diffuse idiopathic skeletal hyperostosis (DISH) is an ankylosing skeletal condition. It was originally described by Forestier et al. in 1950 as calcification of the anterolateral perivertebral ligaments (1). Stiffness and decreased range of motion of the spine, mainly at the cervical level, are the most common clinical presentation of DISH, however, extra-skeletal manifestations may also be present when protrusion of the hyperostotic formations impinges or obstructs anteriorly the pharynx, larynx, trachea or esophagus leading to dysphagia (2). Dysphagia is an uncommon symptom of DISH as it may affect 0.1% to 6% of the adult population at some point in time (3). The affected patient may feel a lump in the throat, have difficulties for swallowing solid foods or even liquid foods (4). Physical examination should include, in addition to a classical spinal neurological examination, thorough assessment of the neck, mouth, oropharynx and larynx by Ear, Nose and Throat (ENT) surgeons. DISH diagnosis can be established on cervical spine radiographs and computed tomography (CT) scan and its implication in dysphagia can be confirmed on Barium swallow fluoroscopy (2). Barium swallow fluoroscopy represents a key investigation in accurately identifying the level of dysphagia which could help in eventually guiding surgical management if required (5). Conservative approach is usually the first line of management consisting of soft diet associated to nonsteroidal anti-inflammatory drugs (NSAIDs) and swallow therapy, however, if the patient’s symptoms do not improve or worsen, surgical management by anterior cervical osteophytectomy (Smith Robinson approach) represents the second line of management and is very effective in relieving the symptomatic dysphagia (5,6). Rarely anterior cervical fusion may be required in case of combined neurological and extra-skeletal manifestations (7). Identifying the symptomatic level is crucial as targeting specifically one level would obviate the need of a wide cervical soft tissue dissection and decrease intraoperative and postoperative risks. However, there is a lack of consensus on whether to remove only the most important osteophyte or to include all DISH levels, which could impact surgical outcomes.

The case of a 51-year-old patient is presented; he has been complaining of throat discomfort and dysphagia for several months. Examination and investigations confirmed dysphagia originating from DISH. Surgical management and its steps are illustrated and the important role of Barium swallow fluoroscopy in identifying the target level and confirming satisfactory postoperative outcome is shown, which has not been previously demonstrated in the literature. We present this case in accordance with the CARE reporting checklist (available at https://jss.amegroups.com/article/view/10.21037/jss-24-84/rc).

Case presentation

All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Helsinki Declaration (as revised in 2013). Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the editorial office of this journal.

A 51-year-old man, with no specific past medical history, presented to the ENT clinic because of a 3-month pain in the left side of the tongue base with sensation of a lump in the throat and dysphagia that has been steadily increasing. No neck stiffness was noted on physical examination. Fiberscope examination showed tender swelling at the tongue base on the left. CT scan of the neck revealed DISH extending from C4 to C7 with major osteophytes mainly at C4–C5 (Figure 1A,1B), and barium swallow fluoroscopy showed asymmetrical opacification with esophagus indentation mainly on the left side at C4–C5 (Figure 2). After failure of conservative treatment for 6 weeks consisting of soft diet and anti-inflammatory medications, the patient underwent surgery at Kingdom Hospital (Riyadh, Saudi Arabia, Anouar Bourghli’s former hospital) consisting of resection of the osteophyte (osteophytectomy) at C4–C5 being the only pathological level confirmed on Barium swallow fluoroscopy. Through an anterior cervical pre-vascular approach, high-speed burr was used to gradually remove the osteophytes leaving a flat bony surface (Figure 3A,3B) and intraoperative X-rays confirmed satisfactory resection (Figure 4A,4B). Meticulous hemostasis was achieved using bone wax to avoid postoperative hematoma in the surgical bed. Anterior cervical decompression or fixation were not required as the patient did not have any radicular or myelopathic complaints. Difficulty in swallowing improved gradually, 3 months after the surgery he had no evidence of dysphagia as confirmed on barium swallow fluoroscopy showing a wide esophageal lumen at C4–C5 (Figure 5).

Figure 1 Computed tomography scan confirming the diagnosis of diffuse idiopathic skeletal hyperostosis (DISH) extending from C4 to C7 with major osteophytes mainly at C4–C5 (A,B).

Figure 2 Barium swallow fluoroscopy showing asymmetrical opacification with esophagus indentation mainly on the left side at the level of C4–C5 (black arrow).

Figure 3 Intraoperative picture depicting the resection of the osteophyte at C4–C5 with the use of a high-speed burr (A) with a remaining flat bony surface at the end of the procedure (B). L, left; R, right; P, proximal; D, distal.

Figure 4 Intraoperative lateral X-ray before the osteophyte resection (A) with confirmation of a postoperative satisfactory outcome (B).

Figure 5 Three months postoperative barium swallow fluoroscopy confirming symmetrical opacification with wide esophageal lumen at C4–C5.

Discussion

DISH is a systemic condition of unknown etiology with a prevalence of 8–10% in patients aged over 65 years and rarely seen in those aged under 45 years (8). It is characterized by 3 criteria identified by Resnick et al. (8): calcification and ossification along the anterolateral paravertebral ligaments of at least 4 vertebral bodies, relative preservation of the intervertebral discs height and absence of ankylosis in the interapophyseal joints. Exuberant osteophyte formations resulting from ossification of the anterior longitudinal ligament may lead to otolaryngological symptoms such as sore throat, swallowing difficulty (dysphagia) or dysphonia and there is no association between osteophyte size and symptoms (9-11). Dysphagia is the most frequently reported extra-skeletal manifestation of DISH and occurs in about 30% of patients with cervical osteophytes (7).

Pathophysiology for the occurrence of dysphagia after DISH may involve several mechanisms such as luminal impingement of the esophagus by a large osteophyte, irritation by the osteophyte leading to inflammation and edema around the esophagus, pain and muscle spasm, or combination of any of the aforementioned mechanisms (4,12). A meta-analysis of anterior cervical hyperostosis related dysphagia found that the main duration of secondary symptoms before diagnosis of cervical DISH was 500 days (ranging from 1 to 3,010 days) which underlines the chronicity of the symptoms in some cases, the difficulty in diagnosing the symptoms associated with DISH and prolonged implementation of conservative treatments (13). Dysphagia may occur with both solids and liquids depending on disease progression and cervical location and secondary unintentional weight loss with decline in nutritional status is frequent and should be considered as an alarming event driving the management toward a radical solution (13).

Conservative approach should be the first line of management in case of DISH-related dysphagia consisting of soft diet, NSAIDs and swallow therapy for 6 weeks. However, if the patient’s symptoms do not improve or worsen gradually over time, surgical management should be considered the treatment of choice consisting of osteophytectomy at the major levels through and anterior prevascular approach and fusion may occasionally be needed if there are associated neurological symptoms (radiculopathy, myelopathy) (14) or signs of instability (10,15). Patients usually show significant symptomatic improvement within 3 months after the surgery (15).

Many articles in the literature showed the interest of Barium swallow fluoroscopy in confirming the diagnosis of DISH related dysphagia and recognizing the concerned levels (2,15), however, none of them insisted on the fact that the true interest of accurately identifying the compression level should serve as a guide for the surgical management by limiting the osteophytectomy only to the affected level ignoring the remaining non-symptomatic ones despite their eventual big size. In the paper by Lui Jonathan et al. (2), several postop CT scans showed resection of the whole osteophytes from C3 to T1, which implies wider cervical soft tissue dissection and higher intraoperative and postoperative risks including hematoma, infection, recurrent laryngeal nerve palsy, vertebral artery injury and esophageal perforation. In fact, one of the 6 patients of their series suffered from a post-operative hematoma that required evacuation and admission to the intensive care unit, such event might have been caused by the fact that osteophytectomies at multiple levels would lead to increased bone bleeding and subsequent hematoma (2).

In the case of our patient, osteophytectomy targeted only the C4–C5 level, patient was discharged on Day 1 with quick recovery and in addition to the clinical improvement, the postoperative barium swallow fluoroscopy at 3 months clearly showed dynamic radiological improvement with symmetrical opacification and wide esophageal lumen at C4–C5. The current study presents, nevertheless, several limitations such as being a single case report and lacking long-term follow-up. Future studies including a higher number of patients with long-term follow-ups are warranted in order to confirm such targeted strategy and elaborate proper guidelines and clear consensus for the surgical management of DISH related dysphagia.

Conclusions

The current case thoroughly illustrated the diagnosis and surgical management in the presence of dysphagia from DISH. Through an anterior pre-vascular approach complete resection of the major osteophytes could be done. Barium swallow fluoroscopy showed very high interest in dynamically demonstrating the main level causing the dysphagia symptoms and also confirming satisfactory esophagus decompression and release after surgery.

Acknowledgments

Funding: None.

Footnote

Reporting Checklist: The authors have completed the CARE reporting checklist. Available at https://jss.amegroups.com/article/view/10.21037/jss-24-84/rc

Peer Review File: Available at https://jss.amegroups.com/article/view/10.21037/jss-24-84/prf

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://jss.amegroups.com/article/view/10.21037/jss-24-84/coif). The authors have no conflicts of interest to declare.

Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. The source of the patient is Kingdom Hospital (Riyadh, Saudi Arabia) which is the former hospital of the first author (Anouar Bourghli) who moved to King Faisal Specialist Hospital and Research Centre (Riyadh, Saudi Arabia). All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Helsinki Declaration (as revised in 2013). Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the editorial office of this journal.

Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.

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