Multifocal recurrence of pleomorphic adenoma of the lacrimal gland

PURPOSE: To report two cases of multifocal recurrent pleomorphic adenoma of the lacrimal gland, and to highlight the clinical and magnetic resonance imaging findings. OBSERVATIONS: The authors present two patients with recurrent pleomorphic adenoma of the lacrimal gland. During their previous primary surgical resection at outside institutions, one patient reportedly had a macroscopically complete excision, while the second patient had violation of the pseudocapsule. Both patients had multiple recurrent nodular lesions detected on magnetic resonance imaging with extension beyond the surgical field of the primary resection. Both underwent subsequent lateral orbitotomy with resection of all identifiable nodules and histopathology was consistent with pleomorphic adenoma. In one patient, two nodules were found two months after the surgery, which grew slowly over the last four years. The second patient had no clinical or radiologic sign of recurrence at last follow up, three years after resection of multinodular recurrence. CONCLUSIONS AND IMPORTANCE: The two cases demonstrate the challenges in the management of multifocal recurrence of lacrimal gland pleomorphic adenoma. The multicentric nature of recurrent lesions in these two cases increase the risk of future recurrence, malignant transformation, and morbidity caused by surgery and radiation. Magnetic resonance imaging is the imaging study of choice, but it may still be inadequate in identifying all the nodules.