The authors report an extremely rare co-occurrence of a thyrotropin secreting pituitary adenoma and a low grade glioma. A 35-year old patient was initially misdiagnosed and treated as a case of primary hyperthyroidism till he presented with visual symptoms and seizures. On evaluation he was also found to have a right frontal glioma. They discuss the presentation, clinical and diagnostic implications, treatment options and possible molecular pathways which help us understand the molecular biology of such tumor associations. They review literature of 12 previous indexed reports of pituitary tumors associated with various grades of gliomas. Though there are 3 previous reports of functioning pituitary tumors and gliomas, this is the first report of a thyrotropin secreting pituitary adenoma and an associated low grade glioma.