In utero virilization secondary to a maternal Krukenberg tumor: case report and review of literature

被引:4
作者
Bustamante, Carmen [2 ]
Hoyos-Martinez, Alfonso [2 ]
Pirela, Daniela [3 ]
Diaz, Alejandro [1 ]
机构
[1] Nicklaus Childrens Hosp, Div Pediat Endocrinol, 3100 SW 62nd Ave, Miami, FL 33155 USA
[2] Nicklaus Childrens Hosp, Dept Med Educ, Miami, FL USA
[3] Mt Sinai Med Ctr, Dept Med Educ, Miami Beach, FL 33140 USA
关键词
disorders of sex development and hyperandrogenism; Krukenberg tumor; PREGNANCY; RECEPTOR; LUTEOMA; BIRTH; OVARY;
D O I
10.1515/jpem-2016-0433
中图分类号
R5 [内科学];
学科分类号
1002 ; 100201 ;
摘要
Background: Krukenberg tumors are ovarian metastatic adenocarcinomas with a primary origin usually located in the stomach, colon, gallbladder, pancreas, or breast. Occasionally, these tumors produce virilization in the affected individual due to androgen production by luteinization of the tumoral stroma. It is believed that during pregnancy these tumors are more likely to increase androgen production due to the elevated levels of human chorionic gonadotropin (hCG). High maternal androgens can cross the placenta producing virilization of the female fetus. Case presentation: A 46, XX newborn female, whose mother was diagnosed with a metastatic ovarian tumor during her second trimester of gestation associated with worsening hirsutism and acne, was found to have ambiguous genitalia at birth. Testosterone levels in both the mother and infant were elevated. Follow-up laboratory tests showed progressive normalization of circulating androgens after delivery. Conclusions: Krukenberg tumors are rare and may produce virilization of the mother and the female fetus when present during pregnancy.
引用
收藏
页码:785 / 790
页数:6
相关论文
共 23 条
[1]  
Al-Agha OM, 2006, ARCH PATHOL LAB MED, V130, P1725
[2]  
BELL RJM, 1977, LANCET, V1, P1162
[3]   Molecular mechanisms of external genitalia development [J].
Blaschko, Sarah D. ;
Cunha, Gerald R. ;
Baskin, Laurence S. .
DIFFERENTIATION, 2012, 84 (03) :261-268
[4]   A systematic review and meta-analysis to revise the Fenton growth chart for preterm infants [J].
Fenton, Tanis R. ;
Kim, Jae H. .
BMC PEDIATRICS, 2013, 13
[5]  
Gardner D.G., 2007, GREENSPANS BASIC CLI, VEighth
[6]  
HARADA N, 1992, AM J HUM GENET, V51, P666
[7]   ADRENAL STEROIDOGENESIS IN VERY-LOW-BIRTH-WEIGHT PRETERM INFANTS [J].
HINGRE, RV ;
GROSS, SJ ;
HINGRE, KS ;
MAYES, DM ;
RICHMAN, RA .
JOURNAL OF CLINICAL ENDOCRINOLOGY & METABOLISM, 1994, 78 (02) :266-270
[8]   Recurrent severe hyperandrogenism during pregnancy: a case report [J].
Holt, HB ;
Medbak, S ;
Kirk, D ;
Guirgis, R ;
Hughes, I ;
Cummings, MH ;
Meeking, DR .
JOURNAL OF CLINICAL PATHOLOGY, 2005, 58 (04) :439-442
[9]   Hyperandrogenic States in Pregnancy [J].
Kanova, N. ;
Bicikova, M. .
PHYSIOLOGICAL RESEARCH, 2011, 60 (02) :243-252
[10]  
Kiyokawa T, 2006, AM J SURG PATHOL, V30, P277