MARC details
| 000 -LEADER |
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| fixed length control field |
03101nam a22003857a 4500 |
| 008 - FIXED-LENGTH DATA ELEMENTS--GENERAL INFORMATION |
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| fixed length control field |
190521s20192019 xxu||||| |||| 00| 0 eng d |
| 022 ## - INTERNATIONAL STANDARD SERIAL NUMBER |
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| International Standard Serial Number |
1750-1172 |
| 024 ## - OTHER STANDARD IDENTIFIER |
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| Standard number or code |
10.1186/s13023-019-1057-x [doi] |
| 024 ## - OTHER STANDARD IDENTIFIER |
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| Standard number or code |
10.1186/s13023-019-1057-x [pii] |
| 040 ## - CATALOGING SOURCE |
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| Original cataloging agency |
Ovid MEDLINE(R) |
| 099 ## - LOCAL FREE-TEXT CALL NUMBER (OCLC) |
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| PMID |
31036049 |
| 245 ## - TITLE STATEMENT |
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| Title |
Gynecologic and reproductive outcomes in fibrous dysplasia/McCune-Albright syndrome. |
| 251 ## - Source |
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| Source |
Orphanet Journal Of Rare Diseases. 14(1):90, 2019 Apr 29. |
| 252 ## - Abbreviated Source |
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| Abbreviated source |
Orphanet J Rare Dis. 14(1):90, 2019 Apr 29. |
| 253 ## - Journal Name |
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| Journal name |
Orphanet journal of rare diseases |
| 260 ## - PUBLICATION, DISTRIBUTION, ETC. |
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| Year |
2019 |
| 260 ## - PUBLICATION, DISTRIBUTION, ETC. |
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| Manufacturer |
FY2019 |
| 265 ## - SOURCE FOR ACQUISITION/SUBSCRIPTION ADDRESS [OBSOLETE] |
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| Publication status |
epublish |
| 266 ## - Date added to catalog |
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| Date added to catalog |
2019-05-21 |
| 501 ## - WITH NOTE |
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| Local holdings |
Available online through MWHC library: 2006 - present |
| 520 ## - SUMMARY, ETC. |
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| Abstract |
BACKGROUND: Autonomous ovarian activation with recurrent estrogen-producing cysts is a hallmark feature of the rare bone and endocrine disorder fibrous dysplasia/McCune-Albright syndrome. Precocious puberty in girls with McCune-Albright syndrome has been well-described, however long-term effects on gynecologic and reproductive function are unknown. Concerningly, case reports have described poor skeletal outcomes associated with pregnancy in women with fibrous dysplasia. |
| 520 ## - SUMMARY, ETC. |
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| Abstract |
CONCLUSIONS: Women with fibrous dysplasia/McCune-Albright syndrome report a high prevalence of gynecologic morbidity and reduced fertility. There is no clear association between pregnancy and poor skeletal outcomes in this population. |
| 520 ## - SUMMARY, ETC. |
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| Abstract |
METHODS: Thirty-nine women with fibrous dysplasia/McCune-Albright syndrome were evaluated as part of a natural history study. Clinical, radiographic, and biochemical data were reviewed. Women were contacted to obtain detailed menstrual and reproductive histories. |
| 520 ## - SUMMARY, ETC. |
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| Abstract |
RESULTS: Abnormal uterine bleeding affected 77% of women (30/39), and was associated with severe anemia requiring blood transfusion in 3 cases. Nine women underwent hysterectomy for management of bleeding, including 67% (6/9) at the unusually young age of less than age 35 years. Infertility affected 43% of women (9/21), including 2 women who developed primary ovarian insufficiency after undergoing surgical treatment of ovarian cysts. Of 25 spontaneous pregnancies in 14 women, 35% (8) were unplanned. Among the 14 pregnancies, pregnancy was associated with no change in bone pain in 7 subjects (53%), increased bone pain in 4 subjects (31%), and decreased bone pain in 2 subjects (15%). No additional skeletal complications were reported during pregnancies. |
| 546 ## - LANGUAGE NOTE |
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| Language note |
English |
| 650 ## - SUBJECT ADDED ENTRY--TOPICAL TERM |
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| Topical term or geographic name entry element |
IN PROCESS -- NOT YET INDEXED |
| 651 ## - SUBJECT ADDED ENTRY--GEOGRAPHIC NAME |
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| Institution |
MedStar Washington Hospital Center |
| 656 ## - INDEX TERM--OCCUPATION |
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| Department |
Obstetrics and Gynecology/Pediatric and Adolescent |
| 657 ## - INDEX TERM--FUNCTION |
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| Medline publication type |
Journal Article |
| 700 ## - ADDED ENTRY--PERSONAL NAME |
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| Local Authors |
Casey, Rachel K |
| 700 ## - ADDED ENTRY--PERSONAL NAME |
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| Local Authors |
Gomez-Lobo, Veronica |
| 790 ## - Authors |
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| All authors |
Boyce AM, Brillante BA, Casey RK, Collins MT, Estrada A, Gomez-Lobo V, Guthrie LC, Murdock CM, Nieman LK, Ovejero Crespo D |
| 856 ## - ELECTRONIC LOCATION AND ACCESS |
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| DOI |
<a href="https://dx.doi.org/10.1186/s13023-019-1057-x">https://dx.doi.org/10.1186/s13023-019-1057-x</a> |
| Public note |
https://dx.doi.org/10.1186/s13023-019-1057-x |
| 942 ## - ADDED ENTRY ELEMENTS (KOHA) |
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| Koha item type |
Journal Article |
| Item type description |
Article |
