[Unique type of Cushing's disease in clinical profile: cyclic Cushing's syndrome and Cushing's disease with favorable outcome to a high daily dose of bromocriptine].

A rare case of cortisol-responsive, dexamethasone-non-responsive cyclic Cushing's disease for whom a high daily dose of bromocriptine was effective was presented, and 58 cases of cushing's syndrome with variable hormonogenesis from the literature were reviewed with our own case (a total of 59 cases). In addition, the results of the high daily dose of bromocriptine treatment in our 7 cases of Cushing's disease were described. The presented case, a 57-year-old female, had a clinically and biochemically proven 3-8 day cycle length. With 40mg daily of bromocriptine, periodic hypercortisolemia disappeared and the patient was in remission with normal suppression of plasma cortisol by dexamethasone. Out of 59 cases of cyclic Cushing's syndrome, 39 were pituitary Cushing's disease or Cushing's syndrome with bilateral adrenal hyperplasia. 15 cases (25%) were adrenal adenoma and ectopic ACTH syndrome. In 22 out of 34 cases (62%), paradoxical response of plasma cortisol to dexamethasone was observed. Only 4 out of 16 cases (25%) on whom Hardy's operation was performed remitted completely. Our own presented case alone showed long-term remission with medical treatment. Seven Cushing's disease patients were treated with a high daily dose of bromocriptine (17.5-55mg/day). Three cases, in whom the highest dose of bromocriptine was 40, 55, and 35mg/day, showed clinical and biochemical long-term remission. In these 3 cases, an escape phenomenon was observed during the early treatment period with a decreased dose of bromocriptine. Thereafter the dose was increased, resulting in a beneficial outcome.(ABSTRACT TRUNCATED AT 250 WORDS)