22963808

PMC3513668

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.

CDC Public Access

Z01 HD008924-01/HD/NICHD NIH HHS/United States
Z01 ND008737-12/ND/ONDIEH CDC HHS/United States
Z99 HD999999/Intramural NIH HHS/United States
ZIA HD008924-01/Intramural NIH HHS/United States