Challenging differential diagnosis, Unusual or unexpected effect of treatment, Rare disease
(Department of Internal Medicine, MidMichigan Medical Center, University of Michigan Health System, Midland, MI, USA)
Am J Case Rep 2015; 16:700-702
Vibratory angioedema (VA) is a rare physical urticaria, with symptoms of itching and swelling of the skin or mucosa when it is exposed to vibration. Avoidance of vibration is the best way to manage this condition. This case report will assist physicians to diagnose this rare condition. Here, a previously unpublished potential successful treatment modality is being presented, with good symptom control, along with some photographs taken during an acute attack. A literature review points towards potential undiagnosed cases.
CASE REPORT: A 70-year-old woman had multiple emergency department visits for tongue and throat swelling over 3 years. The episodes always happened at night. Detailed history elicited some episodes of itching and swelling of hands when driving as well as significant snoring while sleeping. Physical examination was unremarkable except for morbid obesity. Complement factor 4 and C1esterase inhibitor level were within normal limits. A tentative diagnosis of angioedema induced by oropharyngeal vibration from snoring was made. A sleep study confirmed sleep apnea with severe snoring. After CPAP (continuous positive airway pressure) treatment, she had successful symptom control.
CONCLUSIONS: Snoring-induced VA is very likely an under-diagnosed condition in the community. The typical history is the key to the diagnosis. This condition could be confirmed by vibration test or by the resolution of symptoms with elimination of vibration. Effective symptom control is possible by avoidance of oropharyngeal vibration from snoring with the administration of CPAP therapy, making it a potential novel indication for this condition.
Keywords: Angioedema - etiology, Aged, Female, Humans, Snoring - complications, Tomography, X-Ray Computed, Tongue Diseases - etiology, Vibration - adverse effects