Cabergoline treatment for recurrent Cushing’s disease during pregnancy

Abstract

ObJECTIvE: Cushing`s disease during pregnancy is associated with an increased risk for maternal and fetal complications. In recurrent Cushing`s disease following transsphenoidal surgery, and when re-operation is not feasible, medical treatment is usually considered. Cabergoline was found to be effective in reducing hypercortisolism in Cushing's disease. Evolving data concerning the safety of cabergoline use during pregnancy show no significant increase in the rate of complications during pregnancy or the postnatal period. mEThODS: We report a 29-year-old woman, gravida 0, para 0, with recurrent Cushing`s disease, three years after transsphenoidal resection of pituitary aCTh-secreting macroadenoma. Repeated mRI revealed an empty sella with a small gadolinium-enhancing lesion, suspected to be an adenoma remnant on the medial wall of the right cavernous sinus. as the patient was not willing to undergo repeat surgical intervention, treatment with cabergoline was initiated, with a gradual dose titration up to 3.5 mg/week. Clinical improvement ensued, and 4 months later, she conceived spontaneously. after discussing treatment options with the patient, cabergoline treatment at a dose of 2 mg/week was continued throughout pregnancy. RESUlTS: The patient showed complete clinical remission during pregnancy. Consecutive tests of 24-h urinary free cortisol concentration were not found to be elevated. pregnancy and delivery were uneventful except for mild hypothyroidism observed during the second trimester. at full term the patient delivered a healthy female infant, by an elective cesarean section. CONClUSION: This case report demonstrates that cabergoline may be an effective and safe therapeutic option for the treatment of Cushing's disease during pregnancy.Key words: Cabergoline, Cushing's disease, Pregnancy HORMONES 2016, 15(3):453-458 Address for correspondence: Dr. Afif Nakhleh, Endocrinology Department, Bnai Zion Medical Center, 47 Golomb St, Haifa 31048, Israel; Tel.: +972-4-8359931, E-mail: anakhleh@yahoo.com Received: 18-03-2016, Accepted: 26-05-2016 Case report CaSE REpORTA 29 year-old woman, gravida, 0 para 0, was referred to our outpatient clinic with a 3-year history of oligomenorrhea, unexplained weight gain of 20

MANAGEMENT OF ENDOCRINE DISEASE: Management of Cushing’s syndrome during pregnancy: solved and unsolved questions

“…Cabergoline may be an effective and safe therapeutic option for the treatment of Cushing's disease during pregnancy (37,38). Indeed in two reported cases, a pregnancy was obtained while on high-dose cabergoline and maintained throughout pregnancy with complete remission (37,38).…”

Dopamine Agonists for Pituitary Adenomas

“…Surgical resection of the adenoma is the recommended treatment approach; steroidogenesis inhibitors are either contraindicated or indicated only if the risk to the fetus is outweighed by the risks of nontreatment ( 32 ). Case reports suggest that cabergoline 2 mg/week to 5 mg/week might be an option for pregnant women with recurrent disease ( 33 , 34 ), with patients demonstrating UFC normalization and no fetal complications. Of note, one patient required dose escalation up to 5 mg twice weekly to control hypercortisolism by delivery.…”

Pituitary Disease in Pregnancy: Special Aspects of Diagnosis and Treatment?

“…Individual reports have been published describing the use of drugs to treat Cushingʼs disease during pregnancy, particularly the administration of metyrapone 39 . Considering its (limited) efficacy in non-pregnant patients 40 , 41 treatment with the dopamine agonist cabergoline could be tried 42 , 43 . Bilateral adrenalectomy may also represent an option, serving as a means of last resort to manage aggressive, uncontrolled progressive disease during pregnancy 38 , 39 , 44 .…”

“…Zur medikamentösen Therapie des Morbus Cushing in der Schwangerschaft liegen Einzelberichte vor allem zum Einsatz von Metyrapon vor 39 . Vor dem Hintergrund einer begrenzten Wirksamkeit bei nichtschwangeren Patientinnen 40 , 41 kann ein Therapieversuch mit dem Dopaminagonisten Cabergolin sinnvoll sein 42 , 43 . Auch die bilaterale Adrenalektomie kann als Ultima Ratio bei aggressivem, unkontrolliertem Krankheitsverlauf in der Schwangerschaft eine Behandlungsoption darstellen 38 , 39 , 44 .…”