Treatment of aggressive prolactinoma with temozolomide: A case report and review of literature up to date

Abstract

Rationale: Aggressive pituitary adenomas and pituitary carcinomas are rare and demand multiple treatment strategies. Temozolomide, an orally active alkylating chemotherapeutic agent, has recently been recommended as a salvage medication for refractory pituitary adenomas or carcinomas.

Patient concerns: A 17-year-old male presenting with aggressive prolactinoma that continued to progress despite surgery, gamma knife, and dopamine agonists.

Diagnoses: The diagnosis of refractory aggressive prolactinoma was made on the basis of clinical findings and the lack of efficacy of conventional treatment.

Interventions: The patient received the most frequently recommended regimen of temozolomide treatment for 22 cycles.

Outcomes: Temozolomide resulted in a remarkable shrinkage of tumor mass and inhibition of prolactin secretion and this patient's clinical condition improved progressively.

Lessons: Temozolomide can be used as a salvage treatment to refractory pituitary tumors and o(6)-methylguanine-DNA methyltransferase (MGMT) status is a significant predictor to the effectiveness of temozolomide based on the existing literature.

Figure 1

(A) Contrast-enhanced coronal T-1 weighted MRI showed a sellar and suprasellar tumor. (B) Residual tumor 5 months after gamma knife therapy. (C) Sagittal image showed the apparent shrinkage of tumor and the decompressed of optic nerve 7 months after the initiation of cabergoline. (D) Regrowth of tumor mass 26 months after cabergoline. (E) Preoperative MRI demonstrated a 3.5 cm × 3.1 cm × 3.7 cm tumor mass compressing pituitary stalk and optic chiasma. (F) MRI indicated a residual tumor 5 months after operation. (G) MRI indicated that the residual tumor expanded again after second course of gamma knife. (H) Sellar tumor remarkable reduction with cystic degeneration after 22 months of temozolomide treatment.

Figure 2

Graph showing the serum Prolactin concentration [ng/ml] at approximately 1-month interval. Prolactin dropped rapidly after the commencement of temozolomide.