{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,31]],"date-time":"2026-08-31T23:07:05Z","timestamp":1788217625807,"version":"build-2803163510"},"reference-count":33,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2017,8,12]],"date-time":"2017-08-12T00:00:00Z","timestamp":1502496000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/100000054","name":"National Cancer Institute","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000054","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["NCT\u201000027274"],"award-info":[{"award-number":["NCT\u201000027274"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100009633","name":"Eunice Kennedy Shriver National Institute of Child Health and Human Development","doi-asserted-by":"publisher","award":["HHSN261201100018C"],"award-info":[{"award-number":["HHSN261201100018C"]}],"id":[{"id":"10.13039\/100009633","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Pediatric Blood &amp; Cancer"],"published-print":{"date-parts":[[2018,1]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Background<\/jats:title><jats:p>Children with inherited bone marrow failure syndromes (IBMFSs) may be symptomatic in utero, resulting in maternal and fetal problems during the pregnancy. Subsequent pregnancies by their mothers should be considered \u201chigh risk\u201d.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>We retrospectively analyzed outcomes of 575 pregnancies in 165 unaffected mothers of offspring with Fanconi anemia (FA), dyskeratosis congenita (DC), Diamond\u2013Blackfan anemia (DBA), and Shwachman\u2013Diamond syndrome (SDS) for events noted during pregnancy, labor, and delivery. We compared outcomes of pregnancies with affected and unaffected offspring within each group of mothers and with the general population.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>The rates of miscarriage (12\u201320%), elective abortion (5\u201310%), and live birth (68\u201378%) among mothers of all IBMFS groups were similar and comparable with general population rates but recurrent miscarriages (\u22652) were significantly more common in mothers of offspring with DBA and SDS. Offspring with FA were more frequently born small for gestational age (SGA) than unaffected babies (39% vs. 4%) and had fetal malformations (46%) with 18% having three or more, often necessitating early delivery and surgery; offspring with DC had higher rates of SGA (39% vs. 8%) and fetal distress (26% vs. 3%); and offspring with DBA had fetal hypoxia (19% vs. 1%) leading to preterm and emergency cesarean deliveries (26% vs. 6%). Offspring with early\u2010onset severe phenotypes had the most prenatal and peripartum adverse events.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>We identified the high\u2010risk nature of pregnancies in mothers with IBMFS\u2010affected fetuses, suggesting the need for prepregnancy counseling and monitoring of subsequent pregnancies by high\u2010risk fetal\u2013maternal specialists.<\/jats:p><\/jats:sec>","DOI":"10.1002\/pbc.26757","type":"journal-article","created":{"date-parts":[[2017,8,12]],"date-time":"2017-08-12T07:52:47Z","timestamp":1502524367000},"source":"Crossref","is-referenced-by-count":15,"title":["Pregnancy outcomes in mothers of offspring with inherited bone marrow failure syndromes"],"prefix":"10.1002","volume":"65","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7353-1863","authenticated-orcid":false,"given":"Neelam","family":"Giri","sequence":"first","affiliation":[{"name":"Division of Cancer Epidemiology and Genetics Clinical Genetics Branch National Cancer Institute  Rockville Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Helen D.","family":"Reed","sequence":"additional","affiliation":[{"name":"Division of Cancer Epidemiology and Genetics Clinical Genetics Branch National Cancer Institute  Rockville Maryland"},{"name":"Department of Medicine \u2010 Pediatrics Baylor College of Medicine  Houston Texas"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pamela","family":"Stratton","sequence":"additional","affiliation":[{"name":"Office of the Clinical Director Eunice Kennedy Shriver National Institute of Child Health and Human Development National Institutes of Health  Bethesda Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sharon A.","family":"Savage","sequence":"additional","affiliation":[{"name":"Division of Cancer Epidemiology and Genetics Clinical Genetics Branch National Cancer Institute  Rockville Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Blanche P.","family":"Alter","sequence":"additional","affiliation":[{"name":"Division of Cancer Epidemiology and Genetics Clinical Genetics Branch National Cancer Institute  Rockville Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2017,8,12]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.blre.2010.03.002"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.1111\/bjh.14535"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajh.23429"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1469-0705.2002.00753.x"},{"key":"e_1_2_8_6_1","doi-asserted-by":"publisher","DOI":"10.1002\/pd.1055"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.2006.043869"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.1002\/humu.22784"},{"key":"e_1_2_8_9_1","doi-asserted-by":"publisher","DOI":"10.1111\/bjh.13442"},{"key":"e_1_2_8_10_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2141.2010.08212.x"},{"key":"e_1_2_8_11_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmedgenet-2016-104346"},{"issue":"7","key":"e_1_2_8_12_1","first-page":"1","article-title":"Estimated pregnancy rates and rates of pregnancy outcomes for the United States, 1990\u20132008","volume":"60","author":"Ventura SJ","year":"2012","journal-title":"Natl Vital Stat Rep"},{"issue":"2","key":"e_1_2_8_13_1","first-page":"76","article-title":"Recurrent pregnancy loss: etiology, diagnosis, and therapy","volume":"2","author":"Ford HB","year":"2009","journal-title":"Rev Obstet Gynecol"},{"key":"e_1_2_8_14_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.37637"},{"key":"e_1_2_8_15_1","doi-asserted-by":"publisher","DOI":"10.1016\/B978-0-12-397898-1.00002-5"},{"key":"e_1_2_8_16_1","doi-asserted-by":"publisher","DOI":"10.1002\/pd.2829"},{"key":"e_1_2_8_17_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.38.5.336"},{"key":"e_1_2_8_18_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2141.1991.tb08593.x"},{"key":"e_1_2_8_19_1","doi-asserted-by":"publisher","DOI":"10.3324\/haematol.2010.023929"},{"issue":"4","key":"e_1_2_8_20_1","first-page":"530","article-title":"High\u2010risk pregnancies in Diamond\u2010Blackfan anemia: a survey of 64 pregnancies from the French and German registries","volume":"91","author":"Faivre L","year":"2006","journal-title":"Haematologica"},{"key":"e_1_2_8_21_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2141.1999.01668.x"},{"key":"e_1_2_8_22_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2012-06-439679"},{"key":"e_1_2_8_23_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.celrep.2015.09.038"},{"key":"e_1_2_8_24_1","doi-asserted-by":"publisher","DOI":"10.1038\/sj.onc.1205969"},{"key":"e_1_2_8_25_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.rbmo.2014.05.010"},{"issue":"1","key":"e_1_2_8_26_1","first-page":"16","article-title":"Mendelian genetic causes of the short child born small for gestational age","volume":"29","author":"Chernausek SD.","year":"2006","journal-title":"J Endocrinol Invest"},{"key":"e_1_2_8_27_1","doi-asserted-by":"publisher","DOI":"10.1007\/BF00278700"},{"key":"e_1_2_8_28_1","doi-asserted-by":"publisher","DOI":"10.1080\/15476286.2015.1094596"},{"key":"e_1_2_8_29_1","doi-asserted-by":"publisher","DOI":"10.3324\/haematol.2011.055269"},{"key":"e_1_2_8_30_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.placenta.2009.03.005"},{"key":"e_1_2_8_31_1","doi-asserted-by":"publisher","DOI":"10.3109\/14767058.2010.551149"},{"key":"e_1_2_8_32_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM200001063420102"},{"key":"e_1_2_8_33_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.21550"},{"key":"e_1_2_8_34_1","doi-asserted-by":"publisher","DOI":"10.1111\/jpc.12941"}],"container-title":["Pediatric Blood &amp; Cancer"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fpbc.26757","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/pbc.26757","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,30]],"date-time":"2023-09-30T00:52:18Z","timestamp":1696035138000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/pbc.26757"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2017,8,12]]},"references-count":33,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2018,1]]}},"alternative-id":["10.1002\/pbc.26757"],"URL":"https:\/\/doi.org\/10.1002\/pbc.26757","archive":["Portico"],"relation":{},"ISSN":["1545-5009","1545-5017"],"issn-type":[{"value":"1545-5009","type":"print"},{"value":"1545-5017","type":"electronic"}],"subject":[],"published":{"date-parts":[[2017,8,12]]},"article-number":"e26757"}}