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Laryngeal ulcer with Behcet’s disease
  1. Hiroyuki Yano and
  2. Mitsuyo Kinjo
  1. Division of Rheumatology, Department of Internal Medicine, Okinawa Chubu Hospital, Uruma, Japan
  1. Correspondence to Dr Mitsuyo Kinjo; kinjomitsuyo{at}gmail.com

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Description

A 40-year-old previously healthy man presented with 2 weeks of fever and dysphagia due to painful oral ulcers. He subsequently developed an erythematous tender rash on his trunk and extremities, and genital ulcers.

On arrival, the patient could not vocalise due to pain. His temperature was 38.4°C, and other vital signs were unremarkable. Eyes were slightly injected bilaterally. There was no audible stridor. Multiple ulcerations were present on the lips, tongue, buccal membranes, palate and oropharynx. He had painful pustular red-brown nodules on his face, extremities and back, and also genital ulcers. Laboratory tests showed an elevated C reactive protein (20.31 mg/dL); an erythrocyte sedimentation rate was 90 mm/hour. Herpes simplex virus and syphilis tests were negative. Ophthalmologic examination was consistent with previous uveitis. The pathergy test was positive and a skin biopsy of erythema nodosum showed panniculitis. Fiberoptic laryngoscopy revealed multiple aphthous ulcerations on the epiglottis (figure 1A) and arytenoepiglottidean fold (figure 1B). He was diagnosed with Behcet’s disease. He was treated with topical xylocaine, colchicine 0.5 mg/day and prednisolone 30 mg/day, with improvement in his symptoms.

Figure 1

(A) Aphthous ulcerations on the epiglottis. (B) Aphthous ulcerations on the arytenoepiglottidean fold.

Behcet’s disease is a systemic autoimmune disease that is prevalent along the ‘Old Silk Road’ (eg, the Middle East and East Asia).1 Recurrent oral ulcers are the most frequent manifestation. To establish the diagnosis of Bechet’s disease according to International Study Group for Behcet’s Disease, recurrent oral ulceration no less than three times a year is mandatory and at least two of the following findings are required: recurrent genital ulceration, eye lesions, skin lesions and positive pathergy test.2 In a prior study of northern European patient cohort, 5 out of 15 patients (33%) with Behcet’s disease showed laryngeal changes.3 However, laryngeal involvement in Behcet’s disease has been reported only rarely, with complications that included acute airway obstruction, requiring surgical intervention, due to oedema.4–6 Evaluation of the airway should be considered for cases of Behcet’s disease with dysphagia.

Learning points

  • Dysphagia in Behcet’s disease requires evaluation for the infrequent presentation of laryngeal ulcer.

  • Behcet’s disease with laryngeal involvement may lead to airway obstruction.

Acknowledgments

Dr Rita MGill assisted for language.

References

Footnotes

  • Contributors HY and MK contributed to edit and incorporate data, and wrote the manuscript.

  • Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.

  • Competing interests None declared.

  • Patient consent for publication Obtained.

  • Provenance and peer review Not commissioned; externally peer reviewed.

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