Intractable SUNCT cured after resection of a pituitary microadenoma.
BACKGROUND
SUNCT is a rare primary headache disorder that is associated with activation of the posterior hypothalamus and often poorly responsive to medication. Recently, a relationship between between pituitary microadenoma and SUNCT has been suggested, and reports of both amelioration and exacerbation by dopamine-agonists have been published. These findings suggest a functional role for the hypothalamic-pituitary axis in SUNCT.


METHODS
We report the long-term 4 year follow-up of a 35 year-old patient with a 14-year history of medically and surgically intractable SUNCT who experienced immediate and complete resolution of symptoms after resection of a 6 mm pituitary microadenoma.


RESULTS
This patient was first seen at the age of 28 years with a 10-year history of attacks of right retro-orbital pain satisfying the IHS criteria for SUNCT. Many medical and surgical treatments were attempted without success. An MRI demonstrated a 6 mm microadenoma without compression of surrounding structures. A trial of bromocriptine caused marked exacerbation of his pain. The patient underwent a trans-sphenoidal resection of the pituitary lesion. SUNCT attacks worsened for the first 24h post-operatively, then disappeared. He has been completely headache-free, without medication, for the past 43 months with the last follow-up being January 2006.


CONCLUSIONS
This case emphasizes the relationship between pituitary microadenomas and SUNCT, supports the role of the hypothalamic-pituitary axis in the genesis of SUNCT, and illustrates the importance of careful imaging of the pituitary region in patients with SUNCT.
Leroux
Elizabeth
E
Centre Hospitalier Universitaire de Montréal, Campus Notre-Dame, Montreal, QC, Canada.
Schwedt
Todd J
TJ
Black
David F
DF
Dodick
David W
DW
D000236:Adenoma / Q000150:complications* / Q000601:surgery*; D000328:Adult; D006801:Humans; D007030:Hypothalamo-Hypophyseal System / Q000502:physiology; D008279:Magnetic Resonance Imaging; D008297:Male; D019635:Neurosurgical Procedures; D010911:Pituitary Neoplasms / Q000150:complications* / Q000601:surgery*; D050798:SUNCT Syndrome / Q000209:etiology*