Adnexal masses during pregnancy: diagnosis, treatment, and prognosis.

Adnexal masses are identified in pregnant patients at a rate of 2 to 20 in 1000, approximately 2 to 20 times more frequently than in the age-matched general population. The most common types of adnexal masses in pregnancy requiring surgical management are dermoid cysts (32%), endometriomas (15%), functional cysts (12%), serous cystadenomas (11%), and mucinous cystadenomas (8%). Approximately 2% of adnexal masses in pregnancy are malignant. Although most adnexal masses in pregnancy can be safely observed and approximately 70% spontaneously resolve, a minority of cases warrant surgical intervention because of symptoms, risk of torsion, or suspicion of malignancy. Ultrasound is the mainstay of evaluation of adnexal masses in pregnancy because of accuracy, safety, and availability. Several ultrasound mass scoring systems, including the Sassone, Lerner, International Ovarian Tumor Analysis Simple Rules, and International Ovarian Tumor Analysis Assessment of Different NEoplasias in the adneXa scoring systems have been validated specifically in pregnant populations. Decisions regarding expectant vs surgical management of adnexal masses in pregnancy must balance the risks of torsion or malignancy with the likelihood of spontaneous resolution and the risks of surgery. Laparoscopic surgery is preferred over open surgery when possible because of consistently demonstrated shorter hospital length of stay and less postoperative pain and some data demonstrating shorter operative time, lower blood loss, and lower risks of fetal loss, preterm birth, and low birthweight. The best practices for laparoscopic surgery during pregnancy include left lateral decubitus positioning after the first trimester of pregnancy, port placement with respect to uterine size and pathology location, insufflation pressure of less than 12 to 15 mm Hg, intraoperative maternal capnography, pre- and postoperative fetal heart rate and contraction monitoring, and appropriate mechanical and chemical thromboprophylaxes. Although planning surgery for the second trimester of pregnancy generally affords time for mass resolution while optimizing visualization with regards to uterine size and pathology location, necessary surgery should not be delayed because of gestational age. When performed at a facility with appropriate obstetrical, anesthetic, and neonatal support, adnexal surgery in pregnancy generally results in excellent outcomes for pregnant patients and fetuses.

Testosterone serum levels are not predictive of maternal virilization in hyperreactio luteinalis.

BACKGROUND: Elevated concentrations of circulating testosterone are present in hyperreactio luteinalis (HL), a pregnancy-specific, self-limited condition. HL is associated with maternal virilization in about 30% of cases. The correlation between testosterone levels and maternal virilization has not yet been quantified. Our aim was to identify a testosterone cut-off level which may allow to predict maternal virilization. METHODS: A literature research was performed. Publications were chosen if serum testosterone concentrations and presence or absence of maternal virilization was mentioned. Additionally, we report serial levels of steroids analyzed by Liquid chromatography-tandem mass spectrometry (LC-MS/MS) in one case of HL managed at our institution. RESULTS: In all, 31 cases fulfilled the search criteria. We found significant overlap between testosterone levels in asymptomatic women and women with signs of virilization (range 6.2-37.3 nmol/l and 13.7-197.5 nmol/l, respectively). The method applied for testosterone analysis was mentioned in three reports only. Peak serum testosterone concentration in our case was 120.3 nmol/l. CONCLUSION: From the available data, maternal virilization in HL cannot be predicted by the level of circulating testosterone. However, comparability of results is hampered by the analytical methods applied. LC-MS/MS should preferably be used for reporting concentrations of circulating testosterone.

Hyperreactio luteinalis - two accidental findings during cesarean section.

OBJECTIVE: To present two cases of patients with hyper-reactio luteinalis at the cesarean section. DESIGN: Case report. SETTING: Department of Obstetrics and Gynaecology, Hospital Trenčín; Medirex Group Academy n.o., Bratislava. CASE REPORT: We report two cases of women with preeclampsia who we diagnosed with bilateral multi-cystic ovarian enlargement by chance during cesarean section. At both of them the level of human chorionic gonadotropin was above normal, one of the patients had medical history of ovarian serous borderline tumor and this pregnancy was multiple after in vitro fertilization and embryo transfer. Adnexectomy, resection of ovaries and biopsy were carried out. Histologically hyperreactio luteinalis was confirmed in both patients. CONCLUSION: We discuss the necessity of surgical treat-ment, the authors want to emphasize the need for proper preoperative diagnosis and indication of conservative management of patients with hyperreactio luteinalis.

A case of recurrent hyperreactio luteinalis in three spontaneous pregnancies.

Hyperreactio luteinalis is a rare condition in pregnancy characterized by enlarged ovaries with multiple theca luteal cysts, and recurrence of disease has seldom been documented in the literature. This is a case report of a woman who developed recurrent hyperreactio luteinalis with three spontaneous pregnancies. Endocrine evaluation was performed and revealed hyperandrogenism. Ultrasonography was used to assess the ovaries throughout each pregnancy. The ovarian cysts required drainage in the first pregnancy due to severe distention and shortness of breath. Cyst resolution occurred in the post-partum period following each pregnancy. © 2016 Wiley Periodicals, Inc. J Clin Ultrasound 44:502-505, 2016.

Hyperreactio Luteinalis: Maternal and Fetal Effects.

Hyperreactio luteinalis is a rare condition in which there is massive cystic enlargement of the ovaries, mimicking malignancy, during pregnancy. When confronted with this condition, the fear of missing a cancer diagnosis often leads the physician to react with unnecessary surgical intervention, potentially resulting in impaired future fertility. The literature on the subject contains mainly case reports and one small case series. A recent review attempted to summarize what is currently known, but there has not yet been a pervasive change in the approach to the management of this condition. In order to define the natural history of the condition and its maternal and fetal effects, we examined all case reports available in the English literature from 1993 to 2014, in addition to another as yet unpublished case report. Our analysis suggests that, despite its impressive presentation with ovarian enlargement and hyperandrogenism, hyperreactio luteinalis tends to be self-limiting, with spontaneous postpartum resolution and without untoward maternal or fetal sequelae. In particular, fetal virilization is rare, and dependent on the timing of hyperandrogenism. Adverse pregnancy outcomes are likely a consequence of the abnormally high hCG levels observed in many of these gestations, and the subset of women with these abnormal values should be considered for enhanced surveillance. Vaginal delivery is preferred, and strategies to sustain the potential for breastfeeding must be introduced while maternal androgen levels fall, allowing lactation to be established. Considering its benign nature and postpartum resolution, management of HL must be conservative, and continued education of health care professionals who may encounter this entity is vital.

L'hyperreactio luteinalis est un trouble rare qui donne lieu à une hypertrophie kystique des ovaires de grande envergure, laquelle imite la présence d'une tumeur maligne, pendant la grossesse. La présence d'une telle situation mène souvent le médecin à procéder inutilement à une intervention chirurgicale motivée par la crainte de voir un diagnostic de cancer passer inaperçu, ce qui pourrait affecter la fertilité future de la patiente. La littérature sur le sujet consiste principalement en des exposés de cas et en une série de cas de faible envergure. Bien qu'une récente analyse ait tenté de résumer les données connues à ce sujet à l'heure actuelle, aucune modification n'a encore été universellement apportée à la prise en charge de cette pathologie. Afin de définir l'histoire naturelle de cette dernière et d'en déterminer les effets maternels et fœtaux, nous nous sommes penchés sur tous les exposés de cas ayant été publiés en anglais entre 1993 et 2014, ainsi que sur un exposé de cas n'ayant pas encore été publié. Notre analyse laisse entendre que, malgré sa présentation spectaculaire (hypertrophie ovarienne et hyperandrogénie), l'hyperreactio luteinalis a tendance à être spontanément résolutive à la suite de l'accouchement, sans séquelles indésirables pour la mère ou le fœtus. En particulier, la virilisation fœtale est rare et dépend de la chronologie de l'hyperandrogénie. Les issues de grossesse indésirables sont probablement attribuables aux taux anormalement élevés de hCG qui sont constatés dans bon nombre des grossesses affectées; la mise en œuvre d'une surveillance accrue devrait être envisagée pour ce qui est du sous-groupe des femmes qui présentent de telles valeurs anormales. L'accouchement vaginal est à privilégier et des stratégies visant à soutenir le potentiel d'allaitement doivent être mises en œuvre pendant la chute des taux maternels d'androgènes, afin de permettre la lactation. Compte tenu de la nature bénigne de l'hyperreactio luteinalis et de sa résolution postpartum, la prise en charge de ce trouble doit être conservatrice; de plus, la formation continue des professionnels de la santé qui pourraient devoir y faire face est cruciale.

Hyperreactio luteinalis and follicle-stimulating hormone receptor gene activation mutations: A case report.

INTRODUCTION AND IMPORTANCE: Spontaneous ovarian luteal hyperfunction after pregnancy is associated with activating mutations in the follicle-stimulating hormone receptor gene, and clarification of the etiology can help with subsequent treatment. PRESENTATION OF CASE: A 32-year-old woman presented with enlarged ovaries and bilateral ovarian polycystic echoes at 12 weeks of both pregnancies. The first pregnancy underwent transabdominal bilateral ovarian aspiration at 17 weeks and was spontaneously aborted 4 days after the procedure. After the discovery of bilateral ovarian polycystic echoes in the second pregnancy, genetic testing suggested the presence of activating mutations in the follicle-stimulating hormone receptor (FSHR) gene, resulting in ovarian luteinization, and the patient's condition was stabilized after conservative treatment. CLINICAL DISCUSSION: Ovarian luteal hyperfunction may be associated with hyperandrogenemia, thyroid-stimulating hormone abnormalities, abnormal testosterone levels, and genetic mutations. When ovarian luteal hyperfunction occurs, it is recommended to search for the etiology and treat the symptoms. CONCLUSION: Patients presenting with spontaneous ovarian hyperlutealization should be operated on cautiously, treated conservatively, closely observed, and managed for complications, and genetic testing should be performed to clarify the etiology if necessary.

Hyperreactio luteinalis (enlarged ovaries) during the second and third trimesters of pregnancy: common clinical associations.

The objective of this series was to assess sonographic and clinical findings in patients with hyperreactio luteinalis (HL; enlarged ovaries). We retrospectively identified 31 patients with HL and collected data including gestational age, maximum ovarian size, and pregnancy outcomes. Hyperreactio luteinalis was detected at a mean gestational age of 21.6 weeks, reaching average maximum ovarian volumes of 417 and 359 mL on the right and left, respectively. After the first trimester, HL appears to be associated with multiple gestations, twin-twin transfusion syndrome, gestational trophoblastic disease, and Beckwith-Wiedemann syndrome. When HL is present, maternal complications such as preeclampsia are common, and preterm delivery often results.

Hyperreactio luteinalis associated with fetal hyperandrogenism and cystic hygroma.

A 23-year-old woman with a gestational age of 17 weeks presented with abdominal pain. The ultrasound showed maternal hyperreactio luteinalis with fetal cystic hygroma. After termination of pregnancy, the female fetus showed masculinization features with muscular hypertrophy. The hyperreactio luteinalis regressed under hormonal suppression therapy.

Atypical preeclampsia before 20 weeks of gestation with multicystic placenta, hyperreactio luteinalis, and elevated sFlt-1/PlGF ratio as manifestations of fetal triploidy: A case report.

Preeclampsia is one of the most common as well as most severe complications of pregnancy, characterized by new-onset hypertension and proteinuria or other organ dysfunction. It predominantly occurs after 20 weeks of gestation. Very rarely, it can be triggered earlier in some specific situations. Here we report a case of fetal triploidy presenting as an extraordinarily early-onset preeclampsia. A healthy 36-year-old multiparous woman who had conceived naturally was hospitalized due to acute-onset severe hypertension accompanied by proteinuria at 18 weeks of gestation. Laboratory testing ruled out the presence of underlying maternal disease. Ultrasound findings, including multicystic large placenta and multiple fetal anomalies, strongly suggested fetal triploidy. Maternal ovaries showed hyperreactio luteinalis. The soluble fms-like tyrosine kinase-1/ placental growth factor (sFlt-1/PlGF) ratio was elevated, at 270. Medical abortion was carried out at 19 weeks of gestation; thereafter, her symptoms quickly resolved. Fetal triploidy was confirmed by genetic testing. We should be aware that fetal disorders including triploidy as well as pre-existing maternal diseases can provoke such very early-onset preeclampsia. Fetal ultrasound evaluation is critical and the sFlt-1/PlGF ratio is important for prompt diagnosis and management to prevent adverse maternal outcomes associated with atypical preeclampsia before 20 weeks of gestation.

Rupture of Bilateral Theca Lutein Cysts During Pregnancy: A Case Report.

Hyperreactio luteinalis is a rare condition characterized by the presence of bilateral theca lutein cysts, which occur rarely in a singleton normal pregnancy. Bilateral multicystic ovarian enlargement occurs due to elevated beta-human chorionic gonadotropin. We present a rare case of a 23-year-old primigravida in her tenth week of gestation, presenting with an acute abdomen, with increasing abdominal girth, diagnosed with a rupture of theca lutein cyst, and posted for laparotomy. Theca lutein cyst doesn't affect the course of pregnancy and has a spontaneous regression after delivery. Our case is different due to the occurrence of cysts in a singleton pregnancy and the rupture of these cysts during pregnancy. Unless complicated by torsion, rupture, or hemorrhage, most theca lutein cysts are managed conservatively.

Unrecognized Primary Hypothyroidism As a Possible Cause of Hyperreactio Luteinalis.

Hyperreactio luteinalis (HRL) is characterised by benign enlargement of ovaries in pregnancy associated with hyperandrogenism. A 19-year-old primigravida presented with breathlessness, abdominal distension and vomiting in the thirteenth week of gestation. Abdominal examination revealed distension of abdomen disproportionate to the gestational age. Ultrasound was suggestive of bilaterally enlarged multicystic ovaries with a characteristic "spoke-wheel" pattern and a diagnosis of HRL was made. Laboratory investigations revealed primary hypothyroidism and elevated testosterone. She was initiated on levothyroxine therapy. Her respiratory distress worsened on the third day of admission for which she underwent emergency laparotomy with cyst aspiration. Thyroid function tests normalized within six weeks after the initiation of therapy and remained normal for the remainder of pregnancy. Serum testosterone levels returned to normal six weeks postpartum. The elevated thyroid-stimulating hormone levels could have contributed to development of HRL by cross-reacting with human chorionic gonadotropin and follicle-stimulating hormone receptors. Hyperandrogenism and ovarian enlargement regresses with levothyroxine therapy.

Delayed postpartum regression of theca lutein cysts with maternal virilization: A case report.

Theca lutein cysts are rare, benign lesions responsible for gross cystic enlargement of both ovaries during pregnancy. This condition is also termed hyperreactio luteinalis. Elevated human chorionic gonadotropin (hCG) levels or states of hCG hypersensitivity seem to promote these changes, which in up to 30% of patients produce clinical signs of hyperandrogenism. Given the self-limiting course of theca lutein cysts, which are subject to spontaneous postpartum resolution, conservative treatment is the mainstay of patient management. Described herein is a rare case of theca lutein cysts with maternal virilization that failed to regress by 9 months after childbirth. Surgical intervention was eventually undertaken, necessitated by adnexal torsion.

Hyperreactio luteinalis and hypothyroidism: A case report.

We report a case of hyperreactio luteinalis (HL) and hypothyroidism diagnosed at 13 weeks of pregnancy. A 29-year-old woman with a spontaneous 13-week singleton pregnancy presented with hirsutism and a pelvic mass. Ultrasound showed a viable pregnancy compatible with the gestational age and multiple cystic lesions on both ovaries. Her TSH levels were markedly elevated and diagnostic of hypothyroidism, for which she was treated. Two days after presentation, she had a spontaneous abortion. The products of conception were histologically normal. Follow-up at 3 months showed resolution of the ovarian cysts, and the previously elevated androgen levels were in the normal range. Hyperreactio luteinalis was diagnosed. Her elevated TSH levels may have contributed to hyperreactio luteinalis through the activation of the hCG receptor.

Hyperreactio luteinalis mimicking malignancy during pregnancy with elevated CA-125.

OBJECTIVE: To report a case with benign bilateral ovarian tumor during pregnancy mimicking malignancy. CASE REPORT: A 32-year-old women at 20 weeks gestation with large bilateral adnexal masses found on prenatal ultrasound. The cysts had been growing gradually over the course of the pregnancy. MRI show huge multiple cysts in both ovaries. CA-125 was elevated at 260 U/mL. Due to initial impression was malignancy, an excisional surgery was done. Pathology revealed multiple luteinized granulosa cells with benign nature. CONCLUSION: Hyperreactio luteinalis (HL) is often asymptomatic and discovered incidentally on ultrasound or at the time of cesarean section. As the lesions are self-resolving, management is conservative and surgical intervention is required only in cases with severe complications. HL can be mistaken for malignancy, especially in cases in which the tumor marker CA-125 is elevated, leading to inadvertent surgery.

Incidental Finding Of Hyperreactio Luteinalis during Caesarean Section in Twin Pregnancy.

BACKGROUND: Some benign changes of the ovaries like hyper reaction luteinalis sometimes cannot be differentiated from malignant ones without histological examination. In these cases, surgical intervention sometimes cannot be avoided. Hyperreactio luteinalis is a condition that can occur only in pregnancy. It is characterised by bilateral benign multicystic ovarian enlargement. CASE REPORT: We present a case of misleading intraoperative findings during Cesarean section that ended with ovariectomy. CONCLUSION: During the Caesarean section, some benign masses of the ovaries, like hyper reaction luteinalis, are difficult to differentiate from malignant disease without histological examination, requiring surgical intervention.

Incidentally found hyperreactio luteinalis in pregnancy.

Hyperreactio luteinalis (HL) is a rare pregnancy-related condition in which the ovaries become massively enlarged bilaterally, occupied by multiple benign theca lutein cysts, secondary to increased ovarian stimulation by beta-human chorionic gonadotropin (B-hCG). HL should resolve spontaneously postpartum, however, their occurrence has led some physicians unfamiliar with the natural history of the condition to perform unnecessary ovarian cystectomies or oophorectomies. A healthy 32-year-old woman was incidentally found to have new onset multicystic ovaries on ultrasound at 31 + 3 weeks gestational age, which continued to enlarge, with a maximum volume of ∼448.0 cm3 and ∼323.5 cm3 in right and left ovaries, respectively. She also developed signs and symptoms of hyperandrogenism, and later abdominal pain which ultimately expedited delivery. This paper demonstrates that familiarity with HL as a clinical entity, its typical presentation and natural history, and targeting conservative management is paramount in minimizing iatrogenic harm by obstetricians given the increased use of ultrasound in pregnancy. Patients presenting after the first trimester with bilateral multicystic ovaries with a "spoke wheel" appearance on ultrasound, hyperandrogenism, abnormally elevated B-hCG, or symptoms consistent with elevated B-hCG should prompt a possible diagnosis.

Theca lutein cysts and early onset severe preeclampsia.

Hyperreactio luteinalis (HL) is a rare condition that is characterized by bilateral ovarian enlargement and multiple thin walled cysts. Hypersensitivity of the ovary to circulating human chorionic gonadotropin (hCG) is playing the main role in pathophysiology. HL observed in cases where there is high serum ß-HCG levels, similarly to gestational trophoblastic disease, multiple pregnancies, hydrops fetalis and after fertiliy treatment. Most of HL are self limiting condition and patients are asymptomatic. Differentiation from ovarian malignancies is important. This is a case report of severe preeclampsia prior to 20 weeks gestation due to hyperreactio luteinalis.

Ovarian torsion caused by hyperreactio luteinalis in the third trimester of pregnancy: a case report.

In this case report, a rare case of an adnexal torsion caused by hyperreactio luteinalis (HL) in the third trimester is described, since adnexal torsions are mainly restricted to the first trimester of pregnancy. In an emergency Cesarean section, the patient gave birth to a healthy female baby weighing 3,300 g and we found an enlarged benign multiple luteinized follicular cyst mass in the right adnexum, which led to an adnexal torsion. After detorsion, both ovaries recovered to their normal sizes two months after the intervention.

Hyperreactio luteinalis after delivery: a case report and literature review.

Hyperreactio luteinalis (HL), characterized by bilateral functional multicystic ovarian enlargement during pregnancy and puerperium, mostly occurs in third trimester. As a rare benign disease, HL is usually self-limited. However, some cases are dignosed by unnecessary operation. This is a case report of HL appearing a week after delivery. The patient took a puncture biopsy of ovarian tumor guided by ultrasound to rule out of ovarian malignancy. Without any specific treatment, the disease regressed spontaneously.

Hiperreacción luteínica durante el tercer trimestre del embarazo. Reporte de caso / Hyperreactio luteinalis during the third trimester of pregnancy. A case report

RESUMEN La hiperreacción luteínica es una entidad poco frecuente del embarazo, que se caracteriza por aumento bilateral y funcional de los ovarios con presencia de quistes de paredes finas. Varias hipótesis sugieren que es causada por el aumento de las concentraciones o la sensibilidad ovárica a la gonadotropina coriónica. La mayoría no presenta síntomas o complicaciones del embarazo, y el diagnóstico se realiza en forma accidental por ecografía de rutina. Es una condición benigna rara, suele autolimitarse y se resuelve espontáneamente después del parto. Sin embargo, a veces no pueden ser diferenciados de los tumores malignos de ovario sin un examen histológico. La cirugía está indicada cuando existen complicaciones como torsión ovárica o peritonitis por hemorragia o rotura. Se presenta un caso de hiperreacción luteínica durante el tercer trimestre del embarazo.

ABSTRACT Hyperreactio luteinalis is a rare entity of pregnancy characterized by bilateral and functional enlargement of the ovaries that present thin-walled cysts. Several hypotheses suggest it is caused by increased concentrations or ovarian sensitivity to chorionic gonadotropin. Many cases do not present symptoms or complications during pregnancy, and diagnosis is done accidentally by routine ultrasound. This rare benign condition is usually self-limited and resolves spontaneously following delivery. However, some have to be differentiated from malignant ovarian tumors by histological examination. Surgery is indicated when there are complications such as ovarian torsion or peritonitis due to hemorrhage or rupture. We present a case of hyperreactio luteinalis during the third trimester of pregnancy.