Forestier Disease as a Cause of Dysphagia

Abstract

INTRODUCTION Diffuse idiopathic skeletal hyperostosis (DISH), also known as Forestier disease, is a noninflammatory condition which may be related to underlying metabolic disease. CASE DESCRIPTION A 74-year-old male, with already known metabolic syndrome - dyslipidaemia, type II diabetes and obesity (IMC 32 kg/m2) – presented for chronic mechanical cervicalgia, with progressive decrease of flexion, extension and axial rotation, and episodes of inflammatory pain treated with non-steroidal anti-inflammatory drugs as needed. Besides the decrease in axial mobility, the physical examination did not show any other relevant changes, such as gag reflex, dysphonia (wet voice), halitosis or bolus impaction. Catarina Dantas Soares1, Nuno Madureira2, Daniela Santos-Faria1 1Rheumatology Department, Local Health Unit of Alto Minho, Hospital Conde de Bertiandos, Ponte de Lima, Portugal 2Centre for Rehabilitation Medicine in the Centro-Rovisco Pais Region, Portugal C Dantas Soares, D Santos-Faria

INTRODUCTION

Diffuse idiopathic skeletal hyperostosis (DISH), also known as Forestier disease, is a noninflammatory condition which may be related to underlying metabolic disease. It is characterised by bone formation in the spine and entheses with prevalence rising with age.^1,2

Although DISH is asymptomatic in many individuals, the presence of spinal ossifications can lead to spinal pain, stiffness and loss of motion. If upper cervical involvement occurs additional symptoms such as dysphagia may be observed.^2,3

Mild cervical pain and dysphagia associated with DISH are typically managed with conservative treatment, including dietary measures, physical and swallowing therapy, nonsteroidal anti-inflammatory drugs, and muscle relaxants. In refractory cases, surgical intervention may be necessary for osteophyte removal, yielding positive outcomes.^2,4

Figure 1. Lateral cervical spine x-ray showing anterior bone formation consistent with DISH with mass effect at the laryngopharynx.

CASE DESCRIPTION

A 74-year-old male, with already known metabolic syndrome - dyslipidaemia, type II diabetes and obesity (IMC 32 kg/m²) – presented for chronic mechanical cervicalgia, with progressive decrease of flexion, extension and axial rotation, and episodes of inflammatory pain treated with non-steroidal anti-inflammatory drugs as needed.

He further mentioned a recent onset of odynophagia and oesophageal dysphagia for solids. Besides the decrease in axial mobility, the physical examination did not show any other relevant changes, such as gag reflex, dysphonia (wet voice), halitosis or bolus impaction. Latest endoscopic studies didn’t reveal any structural cause for the dysphagia. A cervical X-ray was required which showed a finding compatible with DISH with exuberant anterior vertebral body osteoproliferation (Figure 1).

Computed tomography (CT) scans of cervical spine can be seen in Figures 2 and 3.

Figure 2. A sagittal Computed Tomographic (CT) scanning cervical spine showing anterior ossification particularly prominent at the level of C3-C4 with indentation on the posterior pharyngeal wall.

Figure 3. Axial CT scan at the C3-C4 level reveals prominent anterior ossification in close proximity to the laryngopharynx. At this level, calcification of the posterior longitudinal ligament is also observed.

CONFLICT OF INTEREST

The authors declare no conflict of interest.

References

Mader R, Verlaan JJ, Buskila D. Diffuse idiopathic skeletal hyperostosis: clinical features and pathogenic mechanisms. Nat Rev Rheumatol 2013;9(12):741-50. doi:10.1038/nrrheum.2013.165

Luo TD, Varacallo M. Diffuse Idiopathic Skeletal Hyperostosis. [Updated 2022 Sep 4]. In: StatPearls [Internet]. Treasure Island (FL): StatPearls Publishing; 2023 Jan.

Giammalva GR, Iacopino DG, Graziano F, Gulì C, Pino MA, Maugeri R. Clinical and radiological features of Forestier's disease presenting with dysphagia. Surg Neurol Int 2018 Nov 28;9:236. doi: 10.4103/sni.sni_223_18.

Harlianto NI, Kuperus JS, Mohamed Hoesein FAA, de Jong PA, de Ru JA, Öner FC et al. Diffuse idiopathic skeletal hyperostosis of the cervical spine causing dysphagia and airway obstruction: an updated systematic review. Spine J 2022 Sep;22(9):1490-503. doi: 10.1016/j.spinee.2022.03.002.