Resolution of dopamine agonist-resistant hyperprolactinemia by hysterectomy: a case report

Abstract

Prolactin-producing uterine leiomyomas are very rare. Although hyperprolactinemia rapidly improves after removal of such leiomyomas, no preoperative diagnostic test has been established for prolactin-producing uterine leiomyomas. A 45-year-old Japanese woman, gravida 3 para 3, was referred to our hospital for further examination of hyperprolactinemia resistant to a dopamine agonist. A pituitary prolactinoma was undetectable by brain magnetic resonance imaging. A bromocriptine loading test revealed an increased serum prolactin concentration after loading. Examination for the detection of an ectopic prolactinoma revealed a 9.0 cm diameter uterine leiomyoma that had measured 6.6 cm in diameter about six months before the first visit to our hospital. The hyperprolactinemia rapidly improved after hysterectomy. A prolactin-producing uterine leiomyoma should be considered as a possible cause of hyperprolactinemia resistant to dopamine agonists. Responsiveness to dopamine agonists; deterioration of hyperprolactinemia may be diagnostic for prolactin-producing uterine leiomyomas, although further research is required.

Uterine Fibroid Causing Hyperprolactinemia and Paradoxical Prolactin Rise with Dopamine Agonist: Case Report and Systematic Review

“…Hsu et al [17] demonstrated prolactin staining in 22/ 49 cases of cervical cancer, supporting the notion that ectopic production and secretion of prolactin may cause hyperprolactinemia, rather than a factor inhibiting dopamine or stimulating pituitary prolactin secretion. The clear clinical and biochemical response to surgery demonstrated in all cases strongly supports that the uterine fibroids were the source of excess prolactin [19][20][21][22]. Furthermore, Sato and colleagues performed uterine vein sampling in a 45-year-old woman with a uterine fibroid who underwent abdominal hysterectomy [22] with results consistent with uterine prolactin secretion.…”

“…The clear clinical and biochemical response to surgery demonstrated in all cases strongly supports that the uterine fibroids were the source of excess prolactin [19][20][21][22]. Furthermore, Sato and colleagues performed uterine vein sampling in a 45-year-old woman with a uterine fibroid who underwent abdominal hysterectomy [22] with results consistent with uterine prolactin secretion.…”

“…In three of the five cases, including the current case, immunostaining for prolactin was performed and was negative [21,22]. However, it is conceivable that small foci of prolactin secreting cells were not seen on the sections sampled.…”

Ectopic hyperprolactinaemia due to a malignant uterine tumor resembling ovarian sex cord tumors (UTROCST)

“…Sato et al . , report the case of a Japanese female patient with refractory hyperprolactinaemia and negative MRI for pituitary neoplasm with symptom resolution following hysterectomy for a leiomyoma [ 33 ]. Sendura et al .…”

The Interplay Between Prolactin and Reproductive System: Focus on Uterine Pathophysiology

“…In a single blind randomized clinical trial in women with symptomatic leiomyoma ( 42 ), treatment with cabergoline 0.5 mg weekly for three months significantly reduced menstrual bleeding and size of both the uterus and the largest myoma as compared to the control group, also relieving symptoms, mainly pain ( 42 ). However, few case reports ( 43 – 45 ) have documented persistence of hyperprolactinemia following treatment with the dopamine agonists bromocriptine and cabergoline in three women with uterine myomas. In all these patients, resistance to dopamine agonists was diagnosed on the basis of lack of PRL normalization after medical treatment, and reversal of hyperprolactinemia was induced only by hysterectomy, suggesting that in selected cases extra-pituitary sources of PRL excess should be considered in women with proven resistance to dopamine agonists ( 43 – 45 ).…”