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Hyperreactio luteinalis, presented as an acute abdomen

2007; Informa; Volume: 86; Issue: 1 Linguagem: Inglês

10.1080/00016340600603197

1600-0412

Nils‐Halvdan Morken, UNN METTE FRIBERG‐OTTERSTAD, Jarl A. Kahn,

Reproductive Biology and Fertility

Acta Obstetricia et Gynecologica ScandinavicaVolume 86, Issue 1 p. 104-106 Free Access Hyperreactio luteinalis, presented as an acute abdomen NILS-HALVDAN MORKEN, Corresponding Author NILS-HALVDAN MORKEN From the Department of Obstetrics and Gynecology, Telemark Hospital, Skien, Norway: Nils-Halvdan Morken, Department of Obstetrics and Gynecology, Telemark Hospital, 3710, Skien, Norway [email protected]Search for more papers by this authorUNN METTE FRIBERG-OTTERSTAD, UNN METTE FRIBERG-OTTERSTAD From the Department of Obstetrics and Gynecology, Telemark Hospital, Skien, NorwaySearch for more papers by this authorJARL A. KAHN, JARL A. KAHN From the Department of Obstetrics and Gynecology, Telemark Hospital, Skien, NorwaySearch for more papers by this author NILS-HALVDAN MORKEN, Corresponding Author NILS-HALVDAN MORKEN From the Department of Obstetrics and Gynecology, Telemark Hospital, Skien, Norway: Nils-Halvdan Morken, Department of Obstetrics and Gynecology, Telemark Hospital, 3710, Skien, Norway [email protected]Search for more papers by this authorUNN METTE FRIBERG-OTTERSTAD, UNN METTE FRIBERG-OTTERSTAD From the Department of Obstetrics and Gynecology, Telemark Hospital, Skien, NorwaySearch for more papers by this authorJARL A. KAHN, JARL A. KAHN From the Department of Obstetrics and Gynecology, Telemark Hospital, Skien, NorwaySearch for more papers by this author First published: 31 December 2010 https://doi.org/10.1080/00016340600603197Citations: 10AboutSectionsPDF ToolsRequest permissionExport citationAdd to favoritesTrack citation ShareShare Give accessShare full text accessShare full-text accessPlease review our Terms and Conditions of Use and check box below to share full-text version of article.I have read and accept the Wiley Online Library Terms and Conditions of UseShareable LinkUse the link below to share a full-text version of this article with your friends and colleagues. Learn more.Copy URL Hyperreactio luteinalis (HL) is a rare and benign disorder in pregnancy. It is caused by multiple benign cysts originating from the theca granulosa luteinized complex and resulting in marked cystic enlargement of the ovaries. Hyperreactio luteinalis is associated with choriocarcinoma, hydatidiform mole, fetal hydrops and multiple gestations. Association with hyperemesis gravidarum, hyperthyroidism and intra-uterine growth retardation has recently been described (1). In 60% of cases, no predisposing factors are present (2). We report a case of HL presented as an acute abdomen. Case report A healthy 32-year-old woman, gravida 1, para 0, was admitted to the obstetric department at 32 weeks of gestation under a picture of acute abdomen. The abdominal pain started half an hour before admittance. She had a naturally conceived and normally developing duplex pregnancy. The patient complained of severe abdominal pain, but she was not contracting. The abdominal tension was increased. A severe vaginal bleeding was evident at examination. The signs and symptoms raised a strong suspicion of abruption of the placenta. An abdominal ultrasound was performed and revealed viable intrauterine twins. An acute cesarean section (CS) was performed. During the CS, scarce blood-stained fluid was evident in the peritoneal cavity. Two healthy female infants of 2240 and 2500 g, respectively, were delivered. The infants had no signs of virilization. Histopathological examination of the placenta could not verify abruption. On inspection of the pelvic organs, two severely enlarged ovaries with rupture sites and scarce persistent bleeding at the surface were found (Figure 1). Both ovaries had a size of 15×10×8 cm. No biopsy or aspiration of the cysts was performed in fear of provoking an uncontrollable bleeding. The CS was otherwise uncomplicated. Figure 1Open in figure viewerPowerPoint Uterus and cystic enlarged ovaries with visible rupture. The endocrine parameters analyzed are presented in Table I. It became evident that the patient during the last weeks prior to admittance had experienced symptoms of virilization such as increased abdominal and facial hair growth. These signs were even evident in the puerperium but faded gradually. There were no signs of clitorimegaly. At 12 weeks postoperatively the patient had no signs of virilization, and the ovaries appeared with normal size but with multiple follicles visible on vaginal ultrasound. Table I. Endocrine parameters in a patient with hyperreactio luteinalis measured on the first and twelfth day after cesarean section and 8 weeks later. Endocrine parameter (reference value in nonpregnant) First postoperative day Twelfth postoperative day 8 weeks postoperatively s-T (0.3-2.8 nmol/L) 33.9 10.9 1.3 s-A (<6.6 nmol/L) 59.2 * 5.0 sHBG (30–90 nmol/L) * 178 19 s-hCG (<5 IU/L) 38030 296 <5 s-T, serum testosterone; s-A, serum androstendione; sHBG, serum hormone binding globuline; s-hCG, serum human chorionic gonadotrophin; nmol/L, nanomole per litre; IU/L, international units per litre. *Not measured. Discussion The cause of HL is unknown. Its association with conditions of increased serum human chorionic gonadotrophin (hCG) (3) like those mentioned previously has led to speculations that this hormone plays a crucial role. Human chorionic gonadotrophin is important and necessary to rescue the corpus luteum and peaks in the first trimester. Elevations later in pregnancy in otherwise normal singleton pregnancies are rare but has been described in connection with HL (3). This indicates that abnormally elevated serum hCG may be a mechanism for the development of HL. The occurrence of the condition in cases with normal levels of serum hCG may indicate that increased sensitivity of the ovaries to this hormone may also be a possible mechanism for HL. It has been suggested that mutation in the gene for the serum hCG receptor, resulting in increased sensitivity may be the explanation (1). Follicle-stimulating hormone might be another critical factor for the development of HL. This has been contradicted (4). The macroscopic appearance of HL in the ovaries resembles the small cystic phenomena seen in cases of hydatidiform mole in the uterine cavity. It is based on this tempting to speculate whether HL may be an ovarian form of hydatidiform mole and share a common mechanism. Ovarian hyperstimulation syndrome (OHSS) seen in connection with assisted reproduction has been called an iatrogenic form of HL (2). It is characterized by high concentrations of serum estradiol and nearly always instituted by exogenous hCG. In OHSS, high levels of serum vascular endothelial growth factor has been found and linked to the pathogenesis of this disorder. There are even reports speculating on whether the same factor might play a role in the pathogenesis of HL as well (5). In clinical sense, these two lesions may be related phenomena (6). Androgens are produced in the enlarged ovaries in HL. The patient had considerably elevated serum concentrations of testosterone and androstenedione shortly after delivery. The levels normalized postpartum. It is reported that the level may stay high for weeks. In our patient, normal serum concentrations were present at 8 weeks postpartum. Foulk et al. (7) reported that 14–25% of these patients experienced virilization. In contrast, fetal virilization has not been reported. This may be a result of the large capacity of the placenta to aromatize maternal androgens. The typical appearance, the endocrinological values postoperative and the regress of the lesion in the puerperium makes the clinical diagnosis of HL certain, although histopathological examination was not done. A close follow-up of the patient was necessary, as a malignant causality could not for sure be ruled out. It is important for obstetricians to be aware of this benign lesion so that unnecessary resection of pelvic organs is avoided even in cases presented as an acute abdomen. References 1 Gherman RB, Mestman JH, Satin AJ, Goodwin TM. Intractable hyperemesis gravidarum, transient hyperthyroidism and intrauterine growth restriction associated with hyperreactio luteinalis. A case report. J Reprod Med 2003; 48: 553– 6 2 Joshi R, Dunaif A. Ovarian disorders of pregnancy. Endocrinol Metab Clin North Am 1995; 24: 153– 69 3 Bidus MA, Ries A, Magann EF, Martin JN. Markedly elevated beta-hCG levels in a normal singleton gestation with hyperreactio luteinalis. Obstet Gynecol 2002; 99: 958– 61 4 Check JH, Choe JK, Nazari A. Hyperreactio luteinalis despite the absence of a corpus luteum and suppressed serum follicle stimulating concentrations in a triplet pregnancy. Hum Reprod 2000; 15: 1043– 5 5 Takeda T, Minekawa R, Makino M, Sugiyama T, Murata Y, Suehara N. Hyperreactio luteinalis associated with severe twin-to-twin transfusion syndrome. Gynecol Obstet Invest 2002; 53: 243– 6 6 Tanaka Y, Yanagihara T, Ueta M, et al. Naturally conceived twin pregnancy with hyperreactio luteinalis, causing hyperandrogenism and maternal virilization. Acta Obstet Gynecol Scand 2001; 80: 277– 8 7 Foulk RA, Martin MC, Jerkins GL, Laros RK. Hyperreactio luteinalis differentiated from severe ovarian hyperstimulation syndrome in a spontaneously conceived pregnancy. Am J Obstet Gynecol 1997; 176: 1300– 2, discussion 1302–4 Citing Literature Volume86, Issue1January 2007Pages 104-106 FiguresReferencesRelatedInformation