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Ectopic hyperprolactinaemia due to a malignant Uterine Tumor Resembling Ovarian Sex Cord Tumors (UTROCST)
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Dimitriadis, Georgios K., Wajman, Delane, Bidmead, John, Diaz-Cano, Salvador J., Arshad, Sobia, Bakhit, Mohamed, Lewis, Dylan and Aylwin, Simon J. B. (2020) Ectopic hyperprolactinaemia due to a malignant Uterine Tumor Resembling Ovarian Sex Cord Tumors (UTROCST). Pituitary, 23 . pp. 641-647. doi:10.1007/s11102-020-01070-y
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Official URL: https://doi.org/10.1007/s11102-020-01070-y
Abstract
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.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 (10xULN) and (2) normal cranial MRI assuming macroprolactin has been excluded. The majority of extracranial ectopic prolactin-secreting tumors occur in the reproductive organs.
| Item Type: | Journal Article | ||||||||
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| Subjects: | R Medicine > RB Pathology R Medicine > RC Internal medicine R Medicine > RG Gynecology and obstetrics |
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| Divisions: | Faculty of Medicine > Warwick Medical School > Biomedical Sciences Faculty of Medicine > Warwick Medical School |
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| Library of Congress Subject Headings (LCSH): | Hyperprolactinemia, Ectopic pregnancy, Paraneoplastic syndromes, Prolactinoma | ||||||||
| Journal or Publication Title: | Pituitary | ||||||||
| Publisher: | Springer | ||||||||
| ISSN: | 1386-341X | ||||||||
| Official Date: | December 2020 | ||||||||
| Dates: |
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| Volume: | 23 | ||||||||
| Page Range: | pp. 641-647 | ||||||||
| DOI: | 10.1007/s11102-020-01070-y | ||||||||
| Status: | Peer Reviewed | ||||||||
| Publication Status: | Published | ||||||||
| Publisher Statement: | This is a post-peer-review, pre-copyedit version of an article published in Pituitary. The final authenticated version is available online at: http://dx.doi.org/[insert DOI] | ||||||||
| Access rights to Published version: | Open Access | ||||||||
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