1

Introduction

Gluten-related disorders have been known for many years and are an example of an auto-immune disease caused by polymorphism of the immune response. Gluten is the trigger of three heterogeneous sets of conditions: 1) immune-mediated enteropathy (celiac disease), 2) wheat allergy, 3) non-celiac gluten sensitivity . These three distinct diseases can induce many different and specific manifestations, mediated by innate and adaptive immune pathways which are not common to all three conditions . Inhalant and food allergies have been linked with Ménière disease (MD) symptoms and recently a relation between gluten sensitivity (GS) and MD symptoms has been proposed .

2

Case-report

We describe the case of a 63-year-old female affected from January 2007 by definite unilateral (right ear) MD according to the American Academy of Otolaryngology-Head and Neck Surgery Committee on Hearing and Aequilibrium (AAO-HNS CHE) criteria : she had a history of three definitive spontaneous episodes of vertigo lasting 20 min or longer, with audiometrically documented sensorineural hearing loss of at least 60 dB HL on one occasion, tinnitus and aural fullness. A right peripheral vestibular lesion was detected upon bithermal caloric test according to the Hallpike technique using Jongkees’ formula.

The patient also suffered from osteoarthritis of the distal finger joints with mucous cysts, Heberden’s nodes and constipation with recurrent abdominal pain.

Skin prick tests were positive to gliadin after 12 h and were negative to the most common inhalants and food (cow milk proteins, ovoalbumin, water extract from wheat flour; tomato, potato, apple, and carrot [fresh foods]). Both specific IgE tests for common allergens and wheat, and screening for celiac disease were negative. Upper gastrointestinal endoscopy and duodenal biopsy were also negative (Marsh 0). In 2006, the patient had already undergone a total colonoscopy for constipation, recurrent abdominal pain and hemorrhoids with negative results.

A possible role of the hypersensitivity to gliadin was hypothesized and the patient agreed to follow a restrictive gluten-free diet, eliminating foods that contain wheat, rye, barley, oats, farro, kamut and their derivatives. After six months, remission from all symptoms was complete and the benefit still persists. In particular, the progression of the arthritis has been arrested as has the formation of mucous cysts and Heberden’s nodules. The subject has had no further episodes of vertigo and the pure-tone threshold has remained unchanged. She also reported that she was no longer suffering from constipation. This optimal condition still persists after almost five years provided the gluten-free diet is respected. Taking into account avoidance of cross-contamination and quantity and frequency of gluten exposure, the gluten-free diet adherence was excellent .

There was only one casual intake of dietary gluten, which caused a rapid relapse of the hand osteoarthritis after only one day. Having followed the gluten-free diet for almost 5 years, the patient decided to try reintroducing gluten into her diet but, one week later, she had a severe episode of vertigo with aural fullness, slight worsening of hearing loss at low frequencies, reappearance of tinnitus and worsening of hand osteoarthritis.

2

Case-report

We describe the case of a 63-year-old female affected from January 2007 by definite unilateral (right ear) MD according to the American Academy of Otolaryngology-Head and Neck Surgery Committee on Hearing and Aequilibrium (AAO-HNS CHE) criteria : she had a history of three definitive spontaneous episodes of vertigo lasting 20 min or longer, with audiometrically documented sensorineural hearing loss of at least 60 dB HL on one occasion, tinnitus and aural fullness. A right peripheral vestibular lesion was detected upon bithermal caloric test according to the Hallpike technique using Jongkees’ formula.

The patient also suffered from osteoarthritis of the distal finger joints with mucous cysts, Heberden’s nodes and constipation with recurrent abdominal pain.

Skin prick tests were positive to gliadin after 12 h and were negative to the most common inhalants and food (cow milk proteins, ovoalbumin, water extract from wheat flour; tomato, potato, apple, and carrot [fresh foods]). Both specific IgE tests for common allergens and wheat, and screening for celiac disease were negative. Upper gastrointestinal endoscopy and duodenal biopsy were also negative (Marsh 0). In 2006, the patient had already undergone a total colonoscopy for constipation, recurrent abdominal pain and hemorrhoids with negative results.

A possible role of the hypersensitivity to gliadin was hypothesized and the patient agreed to follow a restrictive gluten-free diet, eliminating foods that contain wheat, rye, barley, oats, farro, kamut and their derivatives. After six months, remission from all symptoms was complete and the benefit still persists. In particular, the progression of the arthritis has been arrested as has the formation of mucous cysts and Heberden’s nodules. The subject has had no further episodes of vertigo and the pure-tone threshold has remained unchanged. She also reported that she was no longer suffering from constipation. This optimal condition still persists after almost five years provided the gluten-free diet is respected. Taking into account avoidance of cross-contamination and quantity and frequency of gluten exposure, the gluten-free diet adherence was excellent .

There was only one casual intake of dietary gluten, which caused a rapid relapse of the hand osteoarthritis after only one day. Having followed the gluten-free diet for almost 5 years, the patient decided to try reintroducing gluten into her diet but, one week later, she had a severe episode of vertigo with aural fullness, slight worsening of hearing loss at low frequencies, reappearance of tinnitus and worsening of hand osteoarthritis.