ISSN 1470-3947 (print) | ISSN 1479-6848 (online)

Endocrine Abstracts (2011) 25 P256

Hypertrophic pachymeningitis and pituitary pathology: lymphocyctic hypophysitis or cabergoline related fibrosis?

Rajeev Raghavan1, Elizabeth Mallam2, Neil Scolding2,3 & Karin Bradley1,3

1Department of Endocrinology, University Hospitals Bristol NHS Foundation Trust, Bristol Royal Infirmary, Bristol, UK; 2Department of Neurology, North Bristol NHS Trust, Frenchay Hospital, Bristol, UK; 3University of Bristol, Bristol, UK.

Case history: A 51-year-old lady with a background of classical migraine and amenorrhoea conceived successfully via IVF, without prior endocrine assessment. Symptoms during the third trimester of pregnancy led to the diagnosis of a pituitary mass lesion (see Table). Possible differential diagnoses, on subsequent endocrine review, included macroprolactinoma, microprolactinoma with expansion during pregnancy, or lymphocytic hypophysitis.

After 6 years of dopamine agonist therapy, the patient was then admitted with progressive generalised headaches and vomiting (similar to her symptoms during pregnancy and different from her usual migraine). Neurological assessment, including a comprehensive autoimmune workup and gallium scan, were all unremarkable. CSF analysis was normal except for a mildly elevated protein. A repeat MRI demonstrated new diffuse meningeal thickening. Hypertrophic pachymeningitis (patchy widespread inflammatory thickening of the dura mater) was diagnosed.

Time-lineHistoryProlactin (<700 mIU/l)MR imaging
1998Secondary amenorrhoea
2002Fertility clinic/IVF therapy
2002Pregnancy 36 weeks–significant headaches, small left temporal field defectMacroadenoma
2002Rapid resolution of field defect postpartum without intervention
2003Persistent amenorrhoea 6 months postpartum, Cabergoline started3818Macroadenoma unchanged
2004Cabergoline 500 μg/week36Excellent involution of pituitary lesion
02/2008Cabergoline reduced to 250 μg/week33Pituitary appearance unchanged
08/2008Disabling headaches60Pituitary appearance unchanged
01/2009Pachymeningitis diagnosed. Cabergoline weaned and stopped 07/200941Diffuse dura mater thickening. Pituitary appearance unchanged
2009–2010Headaches resolved within weeks of Cabergoline withdrawal599–1990Complete resolution of meningeal thickening. Pituitary appearance unchanged

Conclusion: We report the rare association of pituitary pathology, treated with an ergot-derived dopamine agonist, and hypertrophic pachymeningitis. Withdrawal of the cabergoline (implicated in cardiac valvular fibrosis) coincided with rapid resolution of the clinical and radiological features associated with pachymeningitis. Whilst this could be purely coincidental, a causal link cannot be definitively excluded.

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