OBJECTIVE AND IMPORTANCE: Lymphocytic hypophysitis is being recognized with increasing frequency, but the long-term course is not well known. Recurrence of lymphocytic hypophysitis after a long interval has never been reported.
CLINICAL PRESENTATION: A 53-year-old woman presented with central diabetes insipidus. Magnetic resonance imaging (MRI) revealed an intrasellar lesion. Transsphenoidal biopsy yielded a diagnosis of lymphocytic hypophysitis. Regression of the lesion was confirmed by follow-up MRI. The patient lived normally, with gradual improvement of diabetes insipidus, until she suddenly became aware of a visual defect, which developed into bitemporal hemianopsia 2 years after the biopsy. MRI revealed a large sellar lesion extending to the hypothalamus. However, the adenohypophysial function remained normal and the mild diabetes insipidus continued unchanged.
INTERVENTION: Prompt corticosteroid treatment was remarkably effective. The visual defect disappeared during steroid therapy, and a significant reduction of the lesion was revealed by MRI.
CONCLUSION: It is suggested that long-term follow-up with endocrinological and radiological studies may be necessary in cases of lymphocytic hypophysitis. Recurrent cases should be promptly treated with steroids when a definitive histological diagnosis had been confirmed.