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

Purpose Moderate hyperprolactinaemia (2–5 times upper limit of normal) occurring in a patient with a normal pituitary MRI is generally considered to be due to a lesion below the level of detection of the MRI scanner assuming macroprolactin and stress have been excluded. Most patients with mild-to-mo...

Full description

Saved in:

Bibliographic Details
Published in:	Pituitary Vol. 23; no. 6; pp. 641 - 647
Main Authors:	Dimitriadis, Georgios K., Wajman, Delane S., Bidmead, John, Diaz-Cano, Salvador J., Arshad, Sobia, Bakhit, Mohamed, Lewis, Dylan, Aylwin, Simon J. B.
Format:	Journal Article
Language:	English
Published:	New York Springer US 01-12-2020 Springer Nature B.V
Subjects:	Adult Agonists Dopamine Dopamine Agonists - therapeutic use Endocrinology Female Human Physiology Humans Hyperprolactinemia - drug therapy Hyperprolactinemia - pathology Immunohistochemistry Magnetic Resonance Imaging Medicine Medicine & Public Health Metastases Middle Aged Ovaries Pituitary Pituitary gland Prolactin Prolactinoma - drug therapy Prolactinoma - pathology Reproductive organs Skull Tumors Uterine cancer Uterine Neoplasms - drug therapy Uterine Neoplasms - pathology Uterus Prolactinoma Paraneoplastic Ectopic Hyperprolactinaemia Uterine neoplasm UTROCST Extracranial
Online Access:	Get full text
Tags:	Add Tag No Tags, Be the first to tag this record!

Description
Summary:	Purpose Moderate hyperprolactinaemia (2–5 times upper limit of normal) occurring in a patient with a normal pituitary MRI is generally considered to be due to a lesion below the level of detection of the MRI scanner assuming macroprolactin and stress have been excluded. Most patients with mild-to-moderate hyperprolactinaemia and a normal MRI respond to dopamine agonist therapy. We present the rare case of a patient who had prolactin elevation typical of a prolactin-secreting pituitary macroadenoma,with a normal cranial MRI, and in whom the prolactin rose further with dopamine agonist treatment. Subsequent investigations revealed ectopic hyperprolactinaemia to a uterine tumor resembling ovarian sex cord tumor (UTROSCT) which resolved following tumor resection. Although mostly considered to be benign, the UTROSCT recurred with recurrent hyperprolactinaemia and intraabdominal metastases. Methods We have systematically and critically reviewed existing literature relating to ectopic hyperprolactinaemia in general and UTROCST specifically. Results Fewer than 80 cases of UTROSCTs have been reported globally of which about 23% have shown malignant behaviour. There are fewer than 10 cases of paraneoplastic hyperprolactinaemia originating from uterine neoplasms including one other case of ectopic hyperprolactinaemia to a UTROSCT. Conclusions Our case demonstrates the importance of screening for extracranial hyperprolactinaemia in the context of: (1) substantially raised prolactin (10× ULN) and (2) normal cranial MRI assuming macroprolactin has been excluded. The majority of extracranial ectopic prolactin-secreting tumors occur in the reproductive organs.
Bibliography:	ObjectType-Case Study-2 SourceType-Scholarly Journals-1 ObjectType-Review-5 ObjectType-Feature-4 content type line 23 ObjectType-Report-1 ObjectType-Article-3
ISSN:	1386-341X 1573-7403 1573-7403
DOI:	10.1007/s11102-020-01070-y