Background: Ectopic pituitary adenomas (EPAs) are exceedingly rare neoplasms, comprising about 0.5% of all pituitary adenomas. These are often misdiagnosed radiologically, while the correct diagnosis requires high index of suspicion on pathology and immunohistochemistry analysis. Clinical Case: 62-year-old female presented to the ED with transient unilateral visual loss. She denied orbital pain, headache, or motor or sensory deficit. Following unremarkable ophthalmology evaluation, initial MRI brain was suggestive of pituitary adenoma. MRI of the pituitary with gadolinium contrast showed nonenhancing focus of about 1.5 cm within the posterior sphenoid sinus adjacent to a normal-appearing pituitary gland. Lab assessment showed no pituitary hormone excess or insufficiency. ENT evaluation with repeat MRI brain with contrast showed a T2 heterogeneously enhancing hyperintense lesion in the clivus measuring 2.1 x 1.9 x 1.3 cm (transverse, AP and CC, respectively) with bony thinning/erosion of the sellar floor, the posterior wall of sphenoid sinus and dorsal clivus. Patient underwent transnasal transsphenoidal resection of the tumor with sample submitted as clival chordoma. Pathology report showed a 3.3 x 2.7 x 0.9 cm tumor with immunohistochemistry positive for ACTH, FSH, synaptophysin, chromogranin, S100 and cytokeratin while staining negative for GH, LH, TSH, prolactin and epithelial membrane antigen (EMA). Reticulin stain showed loss of reticulin network and Ki-67 labeling index was 1%. Neurosurgery and ENT teams confirmed no gross manipulation of the pituitary gland had been performed and findings supported a diagnosis of ectopic pituitary adenoma. On 6-month follow-up patient continued to do well clinically and MRI showed normal-appearing pituitary gland with no residual or recurrent tumor. Conclusion: EPA is a rare entity with about 133 cases described in literature. EPAs are generally benign, however up to 79% are functionally active and more likely than their sellar counterparts to secrete multiple pituitary hormones. EPAs arise from remnants of embryonic pituitary tissue in the migratory part of the Rathke’s pouch during early fetal development. The most common locations for EPA are in the sphenoid sinus, clivus, nasopharynx, cavernous sinus and suprasellar space and these are often misdiagnosed on initial radiology as neuroendocrine tumors, sino-nasal carcinomas, clival chordomas or even fungal infections. It is vital to consider EPA in the differential diagnosis of tumors removed from the mentioned anatomic locations as establishing the correct diagnosis helps to avoid the significant morbidity associated with treatments employed for other tumors. Reference: Riccio, L., Donofrio, C.A., Tomacelli, G. et al. Ectopic GH-secreting pituitary adenoma of the clivus: systematic literature review of a challenging tumour. Pituitary 23, 457-466 (2020)

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Introduction Thyroglossal duct (TGD) cyst is a common congenital anomaly. Thyroid cancer rarely occurs in TGD cysts, affecting less than 1% of cysts. Here we present a case of TGD cancer presenting as a central neck mass. Case Presentation 60 y.o female presented to her primary care physician with a neck mass without any compressive symptoms. She had no history of head/neck radiation exposure and no family history of thyroid cancer. She was noted to have a non-tender central neck mass with palpable cervical lymphadenopathy. Neck ultrasound revealed a 5.7 cm multilobulated cystic and solid mass anterior to the thyroid gland. CT scan of the neck revealed a lobulated mixed solid cystic mass at the level of the thyroid cartilage projecting anteriorly, with an abnormal right submandibular lymph node measuring 2.2×1.0 cm. She was evaluated by ENT and underwent resection of the neck mass and the hyoid bone (Sistrunk's procedure). Level 1b lymph node was also removed. Operative findings were concerning for a malignant process. This was confirmed on pathology with findings of TGD papillary thyroid cancer (PTC) and level 1b lymph node demonstrating metastatic PTC with cystic features. She was then referred to endocrine. Thyroid ultrasound revealed multiple bilateral intermediate suspicion nodules. Left inferior thyroid nodule was noted to have high suspicion sonographic features with abnormal right sided level 2 and 3 lymph nodes. She was referred for total thyroidectomy, pathology revealed multifocal PTC with lymph node involvement with extra nodal extension and metastasis to the left sternohyiod muscle, pT2, pN1b, AJCC stage 2. 6-week post-surgical labs revealed TSH of 1.69 mIU/L (ref range 0.40-4.50), thyroglobulin level of 0.8 ng/ml (ref range athyrotic <0.1), with undetectable thyroglobulin antibodies <1 IU/ml (ref range < or = 1 IU/mL). She underwent RAI with 104.6 mci. I-131 Metastatic survey revealed increased uptake in the thyroid bed, otherwise no increased activity elsewhere. Stimulated TSH and Thyroglobulin levels were done but not collected appropriately and therefore not mentioned here. Repeat labs 12 weeks after RAI revealed a TSH of 2.86 mIU/L, Thyroglobulin level of 0.2 ng/ml, and undetectable Thyroglobulin antibody. Conclusion There are no standardized guidelines for management of TGD cancer given the rarity of this condition. FNAB (Fine need aspiration biopsy) may be associated with high false negatives due to inadequate sampling, given the cystic nature of these lesions. In majority of these cases definitive diagnosis of cancer is made after excision of the TGD cyst. Some experts favor a conservative approach via Sistrunk's procedure and pursuing total thyroidectomy if there is high suspicion of thyroid gland involvement as well. Whereas other authors have suggested a more aggressive approach with total thyroidectomy even without evidence of concomitant involvement of the thyroid gland. Presentation: Saturday, June 11, 2022 1:00 p.m. - 3:00 p.m.