Leah Hawkins Bressler1, Lia A Bernardi1, Mallory A Snyder1, Jian-Jun Wei2, Serdar Bulun1. 1. Division of Reproductive Endocrinology and Infertility, Department of Obstetrics and Gynecology, Northwestern University, Feinberg School of Medicine, Chicago, Illinois. 2. Department of Pathology, Department of Obstetrics & Gynecology, Northwestern University, Feinberg School of Medicine, Chicago, Illinois.
Abstract
BACKGROUND: Aberrant progesterone signaling has been demonstrated in mechanistic studies to be a shared common pathway in fibroids and endometriosis. Progesterone receptor modulation with the selective progesterone receptor modulator (SPRM) ulipristal may decrease pain associated with endometriosis. CASE: A 25-year-old nulligravidae with endometriosis-related pelvic pain refractory to medical and surgical intervention was administered 15mg ulipristal every other day for 3 months. Daily pain scores and bleeding diary were recorded and serum chemistries and hormone levels were checked prior to, during, and after treatment. Pre-treatment and surveillance endometrial biopsy specimens were examined for histology and stained for estrogen and progesterone receptor status. During therapy, pain scores decreased to a median of 0 (P<0.05) and the patient became amenorrheic. Surveillance endometrial biopsy demonstrated SPRM-associated endometrial changes that appeared strikingly similar to simple hyperplasia and resolved with ulipristal discontinuation. Immunohistochemical evaluation demonstrated the presence of estrogen and progesterone receptors before and during ulipristal treatment. CONCLUSIONS: Progesterone receptor modulation with ulipristal substantially improved pain symptoms in a patient with treatment-refractory endometriosis. SPRM-associated changes in the endometrium closely mimicked hyperplasia, developed after less than three months of treatment, and resolved after discontinuation of ulipristal and induction of withdrawal bleed.
BACKGROUND: Aberrant progesterone signaling has been demonstrated in mechanistic studies to be a shared common pathway in fibroids and endometriosis. Progesterone receptor modulation with the selective progesterone receptor modulator (SPRM) ulipristal may decrease pain associated with endometriosis. CASE: A 25-year-old nulligravidae with endometriosis-related pelvic pain refractory to medical and surgical intervention was administered 15mg ulipristal every other day for 3 months. Daily pain scores and bleeding diary were recorded and serum chemistries and hormone levels were checked prior to, during, and after treatment. Pre-treatment and surveillance endometrial biopsy specimens were examined for histology and stained for estrogen and progesterone receptor status. During therapy, pain scores decreased to a median of 0 (P<0.05) and the patient became amenorrheic. Surveillance endometrial biopsy demonstrated SPRM-associated endometrial changes that appeared strikingly similar to simple hyperplasia and resolved with ulipristal discontinuation. Immunohistochemical evaluation demonstrated the presence of estrogen and progesterone receptors before and during ulipristal treatment. CONCLUSIONS: Progesterone receptor modulation with ulipristal substantially improved pain symptoms in a patient with treatment-refractory endometriosis. SPRM-associated changes in the endometrium closely mimicked hyperplasia, developed after less than three months of treatment, and resolved after discontinuation of ulipristal and induction of withdrawal bleed.
Authors: Jacques Donnez; Tetyana F Tatarchuk; Philippe Bouchard; Lucian Puscasiu; Nataliya F Zakharenko; Tatiana Ivanova; Gyula Ugocsai; Michal Mara; Manju P Jilla; Elke Bestel; Paul Terrill; Ian Osterloh; Ernest Loumaye Journal: N Engl J Med Date: 2012-02-02 Impact factor: 91.245
Authors: Serdar E Bulun; You-Hong Cheng; Mary Ellen Pavone; Ping Yin; Gonca Imir; Hiroki Utsunomiya; Stephen Thung; Qing Xue; Erica E Marsh; Hideki Tokunaga; Hiroshi Ishikawa; Takeshi Kurita; Emily J Su Journal: Semin Reprod Med Date: 2010-01 Impact factor: 1.303