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Intractable SUNCT Cured After Resection of a Pituitary Microadenoma

Published online by Cambridge University Press:  02 December 2014

Elizabeth Leroux ,
Todd J. Schwedt ,
David F. Black  and
David W. Dodick
Elizabeth Leroux*
Affiliation:
Centre Hospitalier Universitaire de Montréal, Campus Notre-Dame, Montreal, QC, Canada
Todd J. Schwedt
Affiliation:
Mayo Clinic College of Medicine, Scottsdale, Arizona, USA
David F. Black
Affiliation:
Mayo Clinic College of Medicine, Rochester, Minnesota, USA
David W. Dodick
Affiliation:
Mayo Clinic College of Medicine, Scottsdale, Arizona, USA
*
15 Frédéric Hébert, Rouyn-Noranda, Québec, J9X 1T8, Canada
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Abstract

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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.

Conclusion:

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.

Résumé:

RÉSUMÉ:Contexte:

Le SUNCT (Shortlasting Unilateral Nevralgiform Headache with Conjunctival Injection and Tearing) est une forme rare de céphalée primaire qui est associée à une activation de l’hypothalamus postérieur et qui répond souvent mal à la médication. On présume maintenant qu’il existe une relation entre un microadénome pituitaire et le SUNCT. De plus, certaines publications font état d’une amélioration du SUNCT par les agonistes dopaminergiques alors que d’autres signalent une exacerbation. Ces données suggèrent que l’axe hypothalamo-pituitaire joue un rôle dans le SUNCT.

Observation:

Nous rapportons le cas d’un patient âgé de 35 ans qui a été suivi pendant 4 ans pour un SUNCT présent depuis 14 ans et résistant au traitement médical et chirurgical. Le patient a vu ses symptômes disparaître complètement immédiatement après la résection d’un microadénome pituitaire de 6mm.

Résultats:

Ce patient, qui se plaignait depuis 10 ans d’accès de douleur rétro-orbitaires droites satisfaisant aux critères de l’IHS (International Headache Society) pour le SUNCT, a été vu pour la première fois à l’âge de 28 ans. Plusieurs traitements médicaux et chirurgicaux ont été tentés mais sans succès. L’IRM a montré la présence d’un microadénome de 6mm sans compression des structures avoisinantes. Une tentative de traitement par la bromocriptine a exacerbé sa douleur. Le patient a subi une résection trans-sphénoïdale de la lésion pituitaire. Les accès de SUNCT ont été exacerbés pendant les 24 premières heures après la chirurgie puis ont disparu. Il n’a pas eu de céphalée depuis 43 mois, sans médication. La dernière visite de suivi date de janvier 2006.

Conclusion:

Ce cas souligne la relation entre les microadénomes pituitaires et le SUNCT illustrant le rôle de l’axe hypothalamo-pituitaire dans la genèse du SUNCT et l’importance d’une imagerie minutieuse de la région pituitaire chez les patients atteints de SUNCT.

Type
Original Articles
Information
Canadian Journal of Neurological Sciences , Volume 33 , Issue 4 , November 2006 , pp. 411 - 413
Copyright
Copyright © The Canadian Journal of Neurological 2006

References

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