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Long-Standing Oral Aphthae – A Clue to the Diagnosis of Coeliac Disease

Mouth ulcers are one of the most common complaints of the population. These lesions may be manifestations of relatively benign disorders such as recurrent aphthous stomatitis or may represent mucocutaneous markers of a diversity of systemic diseases. Our report alerts to the awareness of oral aphthae as a clue to silent coeliac disease and subclinical dermatitis herpetiformis. A 51-year-old woman presented in our clinic in 1995 with a 30year history of painful and recurrent oral aphthae. They reached a size of up to 1 cm in diameter comparable to major aphthae and were localized in any area of the oral cavity including the palate. They persisted from weeks to months with individual lesions lasting to a maximum of 6 months. The aphthae were refractory to conventional therapy leading to almost continuous ulcer activity (fig. 1). The family history was unremarkable and there were no other signs of Behçet’s syndrome or inflammatory bowel disease. On further specific questioning the patient recalled non-specific symptoms such as occasional headaches, nervousness, difficulty with concentration and forgetfulness, fatigue, sleep disturbances and constant meteorism. Three years ago she had suffered from diarrhoea for about 1 year and her internist diagnosed hyperthyreoidism. Diarrhoea subsided under therapy with thiamazole; the meteorism, however, did not improve. Laboratory tests revealed: ESR 27 mm/h, haemoglobin 11.8 g/dl (normal 12–16); iron 53 Ìg/dl (60–140); ferritin 14 Ìg/l (7–270); folate 5.2 ng/ml (3–17); vitamin B12 1,376 pg/ml under substitution (200–750). Auto-immune screening showed only a low titre of antinuclear antibodies and C3d-IgG circulating immune complexes of 42.0 Ìg/ml (!9); the Waaler-Rose test was positive. IgG antigliadin antibodies (AGA) were 65 U/ml (!25), IgA AGA 79 U/ml (!25), IgA anti-endomysium antibodies (AEA) 1:160 (1: !10) and antireticulin antibodies (ARA) 1:320 (1: !20). A duodenal biopsy specimen further confirmed the diagnosis of coeliac disease (CD) in our patient. HLA-A1, -B8, -DR3 and -DQ2 were positive; molecular subtyping disclosed the HLA-DQB1*0201 allele, present in more than 90% of patients with CD [1–3]. Because of the known association of CD and dermatitis herpetiformis Duhring (DHD) direct immunofluorescence of normal skin from the gluteal region was performed. Although our patient had never had any sign of DHD there was granular deposition of IgA at the dermo-epidermal junction being characteristic of DHD but not of CD without DHD [1, 4]. Direct immunofluorescence of uninvolved oral mucosa was negative. The patient was put on a gluten-free diet. Within 1 month there was complete resolution of the oral ulcerations. Adhering strictly to that diet our patient has now been followed up for 3 years. She has not experienced any further aphthae and her general health has improved as well. Simultaneously, her haematological parameters normalized, all CD-associated antibodies (AGA, AEA, ARA) declined and were undetectable within 10 months. Fig. 1. Single aphtha with inflammatory halo on the tongue.