Download PDF
Z Gastroenterol 2024; 62(12): 2061-2064
DOI: 10.1055/a-2367-8409
Kasuistik

Dysphagia with recurrent severe aspiration due to cervical diffuse idiopathic skeletal hyperostosis (Forestier’s disease)

Dysphagie mit rezidivierender schwerer Aspiration bei diffuser idiopathischer Skeletthyperostose (Morbus Forestier)
Seyed Alireza Shahvaran 
1   Department of Gastroenterology and Infectiology, Helios Kliniken Schwerin, University Campus of Medical School Hamburg, Schwerin, Germany (Ringgold ID: RIN62491)
2   Medical School Hamburg, Hamburg, Germany (Ringgold ID: RIN236368)
,
Martin Kliment
1   Department of Gastroenterology and Infectiology, Helios Kliniken Schwerin, University Campus of Medical School Hamburg, Schwerin, Germany (Ringgold ID: RIN62491)
,
Stefan Prax
1   Department of Gastroenterology and Infectiology, Helios Kliniken Schwerin, University Campus of Medical School Hamburg, Schwerin, Germany (Ringgold ID: RIN62491)
,
Tobias Paul
3   Department of Neurosurgery and Spinal Surgery, Helios Kliniken Schwerin, University Campus of Medical School Hamburg, Schwerin, Germany (Ringgold ID: RIN62491)
,
Oliver Heese
2   Medical School Hamburg, Hamburg, Germany (Ringgold ID: RIN236368)
3   Department of Neurosurgery and Spinal Surgery, Helios Kliniken Schwerin, University Campus of Medical School Hamburg, Schwerin, Germany (Ringgold ID: RIN62491)
,
Daniel Schmitz 
1   Department of Gastroenterology and Infectiology, Helios Kliniken Schwerin, University Campus of Medical School Hamburg, Schwerin, Germany (Ringgold ID: RIN62491)
2   Medical School Hamburg, Hamburg, Germany (Ringgold ID: RIN236368)
› Author Affiliations
› Further Information
Also available at
    Permissions and Reprints

Abstract

Dysphagia is a common symptom in patients presenting to the gastroenterologist. However, extraluminal causes of dysphagia may be missed by endoluminal diagnosis alone. This case report presents a 72-year-old man with slowly progressive dysphagia that occurred with the ingestion of certain solid foods and was intermittently associated with severe aspiration. Esophagogastroduodenoscopy and barium swallow study were both normal. However, a conventional x-ray and computed tomography (CT) scan of the neck revealed diffuse idiopathic skeletal hyperostosis (Forestier's disease) of the anterior cervical spine (C2–C7). Fiber endoscopic evaluation of swallowing (FEES) revealed functionally incomplete emptying of the oral cavity during swallowing, and the patient reported dysphagia score (EAT-10) was 23 (normal < 3). Surgical removal of the hyperostosis (C3–C6) resulted in immediate and sustained improvement in dysphagia at 3 months.

Zusammenfassung

Die Dysphagie ist ein häufiges Symptom von Patienten, welche sich beim Gastroenterologen vorstellen. Extraluminale Ursachen der Dysphagie können jedoch bei alleiniger endoluminaler Diagnostik übersehen werden. Dieser Fallbericht stellt einen 72-jährigen Mann mit langsam progredienten Schluckbeschwerden vor, welche bei Aufnahme bestimmter fester Nahrungsmittel auftraten und intermittierend mit schweren Aspirationen einhergingen. Die Ösophagogastroduodenoskopie und die Röntgen-Breischluckuntersuchung waren unauffällig. Eine konventionelle Röntgenaufnahme sowie eine Computertomographie (CT) des Halses wiesen jedoch eine diffuse idiopathische Skeletthyperostose (Morbus Forestier) der vorderen Halswirbelsäule (C2–C7) nach. Die fiberendoskopische Evaluation des Schluckaktes (FEES) zeigte funktionell eine unvollständige Entleerung der Mundhöhle beim Schlucken und der Score der vom Patienten berichteten Schluckbeschwerden (EAT-10) betrug 23 (normal < 3). Die chirurgische Entfernung der Hyperostose (C3–C6) führte zu einer umgehenden und anhaltenden Besserung der Dysphagie in der dreimonatigem Nachbeobachtung.

Keywords

Swallowing Disorders - Diffuse Idiopathic Skeletal Hyperostosis - Respiratory Aspiration

Schlüsselwörter

Schluckstörung - Diffuse Idiopathische Skelettale Hyperostose (M. Forestier) - Aspiration


Publication History

Received: 09 May 2024

Article published online:
25 September 2024

© 2024. Thieme. All rights reserved.

Georg Thieme Verlag KG
Oswald-Hesse-Straße 50, 70469 Stuttgart, Germany

 

  • References

  • 1 Namasivayam-MacDonald AM, Riquelme LF. Presbyphagia to Dysphagia: Multiple Perspectives and Strategies for Quality Care of Older Adults. Semin Speech Lang 2019; 40 (03) 227-242
    Thieme ConnectPubMedSearch in Google Scholar
  • 2 Wilkinson JM, Codipilly DC, Wilfahrt RP. Dysphagia: Evaluation and Collaborative Management. Am Fam Physician 2021; 103 (02) 97-106
    PubMedSearch in Google Scholar
  • 3 Verlaan JJ. et al. Diffuse idiopathic skeletal hyperostosis of the cervical spine: an underestimated cause of dysphagia and airway obstruction. Spine J 2011; 11 (11) 1058-1067
    CrossrefPubMedSearch in Google Scholar
  • 4 Forestier J, Rotes-Querol J. Senile ankylosing hyperostosis of the spine. Ann Rheum Dis 1950; 9 (04) 321-330
    CrossrefPubMedSearch in Google Scholar
  • 5 Resnick D, Niwayama G. Radiographic and pathologic features of spinal involvement in diffuse idiopathic skeletal hyperostosis (DISH). Radiology 1976; 119 (03) 559-568
    CrossrefPubMedSearch in Google Scholar
  • 6 Harlianto NI. et al. Diffuse idiopathic skeletal hyperostosis of the cervical spine causing dysphagia and airway obstruction: an updated systematic review. Spine J 2022; 22 (09) 1490-1503
    CrossrefPubMedSearch in Google Scholar
  • 7 Mader R. et al. Metabolic syndrome and cardiovascular risk in patients with diffuse idiopathic skeletal hyperostosis. Seminars in Arthritis and Rheumatism 2009;
    CrossrefPubMedSearch in Google Scholar
  • 8 García Callejo FJ. et al. Incidence of diffuse idiopathic skeletal hyperostosis from a model of dysphagia. Acta Otorrinolaringol Esp 2020; 71 (02) 78-82
    PubMedSearch in Google Scholar
  • 9 Abdel-Aziz M, Azab N, Lasheen H. et al. Swallowing disorders among patients with diffuse idiopathic skeletal hyperostosis. Acta Otolaryngol 2017; 137 (06) 623-626
    CrossrefPubMedSearch in Google Scholar
  • 10 Oude Nijhuis RAB, Zaninotto G, Roman S. et al. European guidelines on achalasia: United European Gastroenterology and European Society of Neurogastroenterology and Motility recommendations. United European Gastroenterol J 2020; 8 (01) 13-33
    CrossrefPubMedSearch in Google Scholar
  • 11 Belafsky PC. et al. Validity and reliability of the Eating Assessment Tool (EAT-10). Annals of Otology, Rhinology & Laryngology 2008; 117 (12) 919-924
    CrossrefPubMedSearch in Google Scholar
  • 12 Kraus EM. et al. Validation and Psychometric Properties of the German Version of the SWAL-QOL. Dysphagia 2018; 33 (04) 431-440
    CrossrefPubMedSearch in Google Scholar
  • 13 Liawrungrueang W. et al. Anterior cervical spine surgery for treatment of secondary dysphagia associated with cervical myelopathy in patient with Forestier’s disease. Ann Med Surg 2021; 72: 103120
    CrossrefPubMedSearch in Google Scholar
  • 14 Cherfane P. et al. Otolaryngologic manifestations of diffuse idiopathic skeletal hyperostosis (Forestier’s disease): A systematic review of the literature. Joint Bone Spine 2021; 88 (06) 105218
    CrossrefPubMedSearch in Google Scholar
  • 15 Scholz C. et al. Long-term results after surgical treatment of diffuse idiopathic skeletal hyperostosis (DISH) causing dysphagia. J Clin Neurosci 2019; 67: 151-155
    CrossrefPubMedSearch in Google Scholar
  • 16 Miyamoto K. et al. Postsurgical recurrence of osteophytes causing dysphagia in patients with diffuse idiopathic skeletal hyperostosis. Eur Spine J 2009; 18 (11) 1652-1658
    CrossrefPubMedSearch in Google Scholar