Aromatase inhibitor treatment of menorrhagia and subsequent pregnancy in a patient with familial hyperparathyroidism jaw tumor syndrome

Capsule:
Aromatase inhibitor therapy improved progesterone-resistant menorrhagia in a patient with hyperparathyroidism–jaw tumor syndrome.

Authors:
Erin F. Wolff, M.D., Micah J. Hill, D.O., William F. Simonds, M.D., James H. Segars, M.D.

Volume 98, Issue 6, Pages 1616-1619, December 2012

Abstract:

Objective:
To describe the clinical management of menorrhagia in a woman with hyperparathyroidism-jaw tumor syndrome (HPT-JT).

Design:
Case report.

Setting:
Large translation research hospital.

Patient:
A 26-year-old nulligravid woman with familial HPT-JT presented with life-long menorrhagia resistant to progesterone IUD therapy and a desire for fertility.

Intervention:
Aromatase inhibitor therapy.

Main Outcome Measures:
Clinical response to therapy and pregnancy.

Result:
Imaging demonstrated an enlarged endometrial lining and thickening of the junctional zone. At operative hysteroscopy, multiple atypical endometrial polyp-like lesions filled the entire uterine cavity and were removed. Histologic evaluation demonstrated the lesions to be adenomyomas with an abundance of aromatase expression. Postoperative treatment included an aromatase inhibitor. The patient’s menorrhagia, which had previously been resistant to progesterone IUD therapy, resolved with the aromatase inhibitor. After ten months of this treatment, the aromatase inhibitor was discontinued and a repeat hysteroscopy revealed a markedly improved uterine cavity. The patient subsequently became pregnant on her first natural cycle and delivered a healthy term infant.

Conclusion:
Aromatase inhibitors may represent a novel treatment for benign uterine pathology in HPT-JT.

  • Micah Hill

    We appreciate the opportunity to present this unique case, especially in an issue of the journal with many discussions on the use of aromatase inhibitors. This patient with HPT-JT had the recently described features of the disease that include uterine abnormalities. In this case she had numerous uterine polypoid adenomas with little improvement after surgery and progestin-IUD treatment. Her adenomas were found to have high expression of aromatase and treatment with an aromatase inhibitor caused resolution of her symptoms and she was able to achieve pregnancy immediately after therapy.

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