A high percentage of diagnosed cases of coeliac disease are oligosymptompatic. Various atypical manifestations such as hypertransaminasemia may guide diagnosis when the disease is suspected. We present two cases of coeliac disease, which were diagnosed on the basis of hypertransaminasemia of unknown origin. One of the patients lacked the gastrointestinal symptoms suggestive of disease. We also retrospectively review (January 1990-December 1998) all the cases of coeliac disease diagnosed in our center in order to establish the frequency of liver enzyme alterations in patients with coeliac disease and their evolution on a gluten-free diet. The importance of sprue suspicion in guiding diagnosis in patients with cryptogenic hypertransaminasemia is highlighted as is the need to rule out underlying liver disease in coeliac patients with persistent hypertransaminasemia after withdrawing gluten from the diet.