An unusual cause of obstructing laryngeal edema: Forestier’s disease. Case report and literature review

An unusual cause of obstructing laryngeal edema: Forestier’s disease. Case report and literature review

Authors

  • Paolo Tropiano a:1:{s:5:"en_US";s:21:"University of Perugia";} https://orcid.org/0000-0002-6328-1511
  • Teodosio De Bonis University of Perugia
  • Gerardo Petruzzi IRCCS ReginaElena National Cancer Institute
  • Giulia Zambonini University of Perugia
  • Sauro Giommetti University of Perugia
  • Mario Faralli University of Perugia
  • Giampietro Ricci University of Perugia

Keywords:

Forestier’s disease, laryngeal edema, DISH

Abstract

Forestier’s Disease or Diffuse Idiopathic Skeletal Hyperostosis (DISH) is a systemic condition characterized by the ossification and calcification of ligaments and entheses. DISH is observed on all continents and in all races, but most commonly in men over 50 years of age. Clinical symptoms resulting from DISH are related to altered skeletal biomechanism that may lead to decrease range of motion and painful stiffness of the axial and peripheral locomotor apparatus. When hyperostosis is localized at the spine at the level of glottic plane, it can lean to ENT symptoms including swallowing disorders, dysphonia, rarely dyspnea. The case report proposed refers to a rare DISH clinical presentation with chronic dyspnea. Fiber optic laryngoscopy was performed: a mucous flap of the posterior wall of the sovraglottic larynx reduced the respiratory space, the edematous tissue was aspired below during inspiration. CT with contrast medium was performed and osteophitic process of the anterior longitudinal ligament was diagnosed, in complete absence of neoplastic neoformation. At first conservative treatment was decided and corticosteroid therapy was introduced. He was managed successfully with an anterior cervical osteophytectomy. DISH is not a rare disease, but it is often undiagnosed. This diagnostic hypothesis should be considered in elderly patients presenting with dysphagia, dysphonia, sleep apnea, pharyngeal globus, which are common symptoms in otolaryngology practice. The suspicion must lead to an endoscopic and radiologic investigations. Early diagnosis is important for the initiation of a multidisciplinary approach that will improve the patient’s quality of life.

References

Forestier J, Rotes-Querol J Senile ankylosis hyperstosis of the spine. Ann. Rheum Dis 1950; 9: 321-330.

Resnick, D, Niwayama, G Radiographic and pathologic features of spinal involvement in diffuse idiopathic skeletal hyperostosis (DISH). Radiology 1976; 119: 559-568.

Marks B, Schober E, Swoboda H Diffuse idiopathic skeletal hyperostosis causing obstructing laryngeal edema. Eur Arch Otorhinolaryngol 1998; 255: 256-8.

Mader R, Verlaan JJ, Buskila D Diffuse idiopathic skeletal hyperostosis: clinical features and pathogenic mechanisms. Nat Rev Rheumatol 2013;9: 741-750.

Pulcherio JO, Velasco CM, Machado RS, Souza WN, Menezes DR Forestier's disease and its implications in otolaryngology: literature review. Braz J Otorhinolaryngol 2014; 80:161-166.

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Published

07-06-2022

Issue

Section

Case Reports: General Surgery and Miscellanea

How to Cite

1.
Tropiano P, De Bonis T, Petruzzi G, Zambonini G, Giommetti S, Faralli M, et al. An unusual cause of obstructing laryngeal edema: Forestier’s disease. Case report and literature review. Acta Biomed [Internet]. 2022 Jun. 7 [cited 2024 Jul. 13];93(S1):e2022128. Available from: https://mattioli1885journals.com/index.php/actabiomedica/article/view/12652