Fertil Steril

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.

22963808

PMC3513668

Journal Article

CDC Public Access