Abstract
Invasive giant prolactinomas are a rare type of prolactin-secreting tumors. Most lactotroph adenomas, including giant prolactinomas, consist of the sparsely granulated subtype and respond well to medical therapy with dopamine agonists. Proptosis due to intra-orbital tumor extension and ischemic infarction are two rare complications associated with these tumors.We report a case of a 51-year-old woman with a 30-year history of a macroprolactinoma who was lost to follow-up and returned with severe proptosis, a 10-cm invasive sellar mass on imaging, and markedly elevated serum prolactin levels, consistent with invasive giant prolactinoma. She was initially managed with dopamine agonists followed by palliative debulking of the tumor, which microscopically demonstrated a highly proliferative neoplasm predominantly consisting of sparsely granulated lactotroph adenoma with a minor component of the rare and aggressive acidophil stem cell adenoma subtype. Postoperatively, she developed a large left middle cerebral artery infarct and ultimately died.This case is notable in that it demonstrates the aggressive nature of invasive giant prolactinomas when not treated and highlights two rare findings in patients with this tumor: orbital invasion and ischemic infarct.
Highlights
Pituitary adenomas are benign tumors of the anterior pituitary, and prolactin-producing pituitary adenomas, so-called prolactinomas, are the most common type [1]
Prolactinomas are classified into three subgroups: the common sparsely granulated lactotroph adenoma (SGLA), the rare densely granulated lactotroph adenoma (DGLA), and the exceptionally rare and poorly characterized acidophil stem cell adenoma (ASCA) [2]
The microscopic findings suggest that, over the years that the patient was lost to follow-up, the neoplasm may have de-differentiated from a benign lactotroph adenoma subtype to a more aggressive one; to our knowledge, this has not been reported in the literature
Summary
Pituitary adenomas are benign tumors of the anterior pituitary, and prolactin-producing pituitary adenomas, so-called prolactinomas, are the most common type [1]. CT scan of the head was done and showed a 10 cm × 7 cm × 6 cm sellar mass invading the left orbit, frontotemporal region, insular lobe, cavernous sinus, paranasal sinuses, and nasopharynx, with associated hemorrhagic necrosis, dilation of the right lateral ventricle, 1 cm of left to right midline shift, and mass effect on the brainstem (Figure 2) Her prolactin level was extremely high at 21,113 ng/mL. A postoperative CT scan showed a residual 7.9 cm × 6 cm × 4.4 cm tumor, as well as thin left frontal and parietal subdural and epidural hemorrhage, scattered surgical site pneumocephalus, and 1.4 cm of midline shift, but was otherwise unchanged from prior scans (Figure 4A) Two days postoperatively, she still had not regained the ability to follow commands, and was decreasingly responsive, so MRI was done to evaluate for possible causes of this change in exam. After discussion with her family, the patient was transitioned to hospice and died soon after extubation
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