Ovarian tumor-derived ectopic hyperprolactinemia

Abstract

To describe extreme hyperprolactinemia originating from a pituitary adenoma in the wall of an ovarian dermoid. This is a description of an unusual case and a review of ectopic prolactin production. Ectopic production of prolactin is a rare condition that has been reported in isolated organ system pathology including ovaries. An ovarian dermoid is a benign neoplasm that has the potential for active unregulated endocrine function. Hyperprolactinemia can result from functioning lactotrophs found in ovarian dermoids and can lead to clinical sequelae. Definitive treatment of the condition requires surgical removal of the functioning endocrine tissue. Extreme hyperprolactinemia caused by a pituitary tumor located within a dermoid has not been reported before. We present a case of profound hyperprolactinemia originating from a pituitary adenoma found in the wall of an ovarian dermoid and give a broad overview of the condition and literature. Ectopic prolactin production should always be considered in symptomatic patients found to have elevated serum levels and no findings on brain imaging.

Challenges and pitfalls in the diagnosis of hyperprolactinemia

“…The predominant form in healthy subjects and in patients with prolactinomas is monomeric PRL (molecular mass 23 kDa), while dimeric or big PRL (45)(46)(47)(48)(49)(50)(51)(52)(53)(54)(55)(56)(57)(58)(59)(60), and big-big PRL or macroprolactin (150-170 kDa) correspond to less than 20% of the total PRL (13)(14)(15). When the serum of a patient with hyperprolactinemia contains mostly macroprolactin, the condition is termed macroprolactinemia (16,17).…”

Incidental diagnosis of ectopic adenohypophyseal tissue in an ovarian dermoid cyst: a case report

“…According to the literature [1], the detection of ectopic pituitary tissue within a mature ovarian teratoma is exceedingly rare and different kinds of pituitary cells could be found [2]. In our patient there was hyperplasia of PRL+ cells in the presence of normal hormone levels and in the absence of clinical endocrine dysfunction.…”

“…Elms et al [2] described a 1.5 Â 1.0 cm ectopic active pituitary neoplasm producing PRL within a dermoid cyst. Moreover, Al-Bazzaz et al [3] reported ectopic adenohypophyseal tissue in a dermoid cyst with predominant PRL-immunopositive cells and also GH and ACTHimmunopositive cells.…”

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

“…In non-pathological states, prolactin secretion has been demonstrated in decidua, mammary tissue, ovaries, the male reproductive tract, epithelial cells, the immune system, brain, hair follicles, and skin [3]. Extra-pituitary pathological prolactin secretion has been described from perivascular epithelioid tumors [4,5], ovarian tumors [6][7][8], acute lymphoblastic and myeloid leukemias [9][10][11], colorectal adenocarcinoma [12,13], non-Hodgkin's lymphoma [14], renal cell carcinoma [15], renal epitheloid angiomyolipoma [16], cervical cancer [17], and lung cancer [18].…”