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

Endocrine Abstracts (2019) 63 P249 | DOI: 10.1530/endoabs.63.P249

Spontaneous pregnancy obtained in persistent Cushing Disease treated with Cabergoline: Report of two cases

Almudena Vicente, Enrique Castro, Andrea Cruz, Ana Castro, Sastre Julia & López José

Deparment of Endocrinology. Hospital Virgen de la Salud, Toledo, Spain.

Introduction: Pregnancy is rare in patients with Cushing Disease (CD) owing to ovulatory dysfunction. When surgery is not curative, medical treatment is usually considered. Ketoconazole and mitotane, commonly used medical therapies, have questionable teratogenicity and are not often options in pregnancy. Cabergoline was found to be effective in reducing hypercortisolism in Cushing’s disease. Observational studies suggested that low dose cabergoline treatment at the time of conception and during early pregnancy is not likely to induce an increase in the risk of miscarriage or of fetal malformation.

Aim: To describe two patients who achieved pregnancy after treated with low-dose cabergoline (CAB) for persistent CD.

Case description 1: We report a 33-year-old woman diagnosed with CD at the age of 23. She underwent trans-sphenoidal resection of pituitary ACTH-secreting microadenoma on January 2005. 18 months after surgery, CD persisted and she underwent Gamma Knife radiosurgery (GKRS). Following GKRS, her cortisol levels remained elevated despite no evidence of visible tumour on pituitary MRI. Adjuvant medical therapy was commenced with low ketoconazole doses with successful control of hypercortisolism for the subsequent 9 years. As she was keen for pregnancy, medical treatment was changed to CAB 0.5 mg twice weekly from July 2015, to avoid the potential teratogenic side effects of ketoconazole. The patient subsequently conceived spontaneously in October 2015.

Case description 2: We report a 30-year-old woman diagnosed with CD at the age of 28, with no visible adenoma on magnetic resonance imaging (MRI). Bilateral inferior petrosal sinus sampling (BIPPS) was done and showed a higher IPS/periphery ACTH ratio on the left side as compared to right. A left hemi-hypophysectomy was performed on February 2018, based on BIPSS findings. Surgery was not curative. As she expressed desire to become pregnant, medical treatment with CAB was started on April 2018, with a gradual dose titration up to 3 mg per week. Clinical improvement was observed and the patient conceived spontaneously in September 2018.

Conclusion: These cases reports demonstrates that cabergoline may be an effective and safe therapeutic option for the treatment of persistent CD in women who want pregnancy.

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