Sheri K Palejwala1, Andrew R Conger1,2, Amy A Eisenberg1, Pejman Cohan1, Chester F Griffiths1, Garni Barkhoudarian1, Daniel F Kelly3. 1. Pacific Neuroscience Institute, John Wayne Cancer Institute at Providence's Saint John's Health Center, 2125 Arizona Ave., Santa Monica, CA, 90404, USA. 2. Department of Neurosurgery, Geisinger Health System, Danville, PA, USA. 3. Pacific Neuroscience Institute, John Wayne Cancer Institute at Providence's Saint John's Health Center, 2125 Arizona Ave., Santa Monica, CA, 90404, USA. kellyd@jwci.org.
Abstract
PURPOSE: In most clinical series of Cushing's disease (CD), over 80% of patients are women, many of whom are of reproductive age. The year following pregnancy may be a common time to develop CD. We sought to establish the incidence of CD onset associated with pregnancy. METHODS: A retrospective review was conducted for patients with biochemically-proven CD. Demographics, clinical history, biochemistry, imaging, pathology, and outcomes were reviewed. Pregnancy-associated CD was defined as symptom onset within 1 year of childbirth. RESULTS: Over 10 years, 77 patients including 64 women (84%), with CD underwent endonasal surgery. Of the 64 women, 64% were of reproductive age (15-45 years) at the time of diagnosis, and 11 (27%) met criteria for pregnancy-associated CD. Of these 11 women, median number of pregnancies prior to onset of CD was 2 (range 1-4) compared to zero (range 0-7) for 30 other women with CD onset during reproductive age (p = 0.0024). With an average follow-up of 47 ± 34 months, sustained surgical remission rates for woman with pregnancy-associated CD, other women of reproductive age, and women not of reproductive age were 91%, 80% and 83%, respectively. The average lag-time from symptom onset to diagnosis for women with pregnancy-associated CD was 4 ± 2 years. CONCLUSIONS: In this exploratory study, over one quarter of women of reproductive age with CD appeared to have symptomatic disease onset within 1 year of childbirth. This relatively high rate of pregnancy-associated CD suggests a possible causal relationship related to the stress of pregnancy and pituitary corticotroph hyperactivity in the peripartum period. This possible association suggests a heightened degree of clinical suspicion and biochemical testing for CD may be warranted after childbirth. Further study of this possible link between pregnancy and CD is warranted.
PURPOSE: In most clinical series of Cushing's disease (CD), over 80% of patients are women, many of whom are of reproductive age. The year following pregnancy may be a common time to develop CD. We sought to establish the incidence of CD onset associated with pregnancy. METHODS: A retrospective review was conducted for patients with biochemically-proven CD. Demographics, clinical history, biochemistry, imaging, pathology, and outcomes were reviewed. Pregnancy-associated CD was defined as symptom onset within 1 year of childbirth. RESULTS: Over 10 years, 77 patients including 64 women (84%), with CD underwent endonasal surgery. Of the 64 women, 64% were of reproductive age (15-45 years) at the time of diagnosis, and 11 (27%) met criteria for pregnancy-associated CD. Of these 11 women, median number of pregnancies prior to onset of CD was 2 (range 1-4) compared to zero (range 0-7) for 30 other women with CD onset during reproductive age (p = 0.0024). With an average follow-up of 47 ± 34 months, sustained surgical remission rates for woman with pregnancy-associated CD, other women of reproductive age, and women not of reproductive age were 91%, 80% and 83%, respectively. The average lag-time from symptom onset to diagnosis for women with pregnancy-associated CD was 4 ± 2 years. CONCLUSIONS: In this exploratory study, over one quarter of women of reproductive age with CD appeared to have symptomatic disease onset within 1 year of childbirth. This relatively high rate of pregnancy-associated CD suggests a possible causal relationship related to the stress of pregnancy and pituitary corticotroph hyperactivity in the peripartum period. This possible association suggests a heightened degree of clinical suspicion and biochemical testing for CD may be warranted after childbirth. Further study of this possible link between pregnancy and CD is warranted.
Authors: G Arnaldi; A Angeli; A B Atkinson; X Bertagna; F Cavagnini; G P Chrousos; G A Fava; J W Findling; R C Gaillard; A B Grossman; B Kola; A Lacroix; T Mancini; F Mantero; J Newell-Price; L K Nieman; N Sonino; M L Vance; A Giustina; M Boscaro Journal: J Clin Endocrinol Metab Date: 2003-12 Impact factor: 5.958
Authors: Bjorn Lobo; Xin Zhang; Garni Barkhoudarian; Chester F Griffiths; Daniel F Kelly Journal: Neurosurg Clin N Am Date: 2015-05-07 Impact factor: 2.509
Authors: Gary D Hammer; J Blake Tyrrell; Kathleen R Lamborn; Carol B Applebury; Elizabeth T Hannegan; Scott Bell; Riva Rahl; Amy Lu; Charles B Wilson Journal: J Clin Endocrinol Metab Date: 2004-12 Impact factor: 5.958