Hyperreactio Luteinalis Research Articles

Conservative laparoscopic surgery for hyperreactio luteinalis after miscarriage of a partial hydatidiform mole coexistent with a fetus: A case report

Conservative laparoscopic surgery for hyperreactio luteinalis after miscarriage of a partial hydatidiform mole coexistent with a fetus: A case report

Ovarian Hyperstimulation and Maternal Virilisation with Successful Pregnancy Outcome

Hyperreactio luteinalis (HL) and ovarian hyperstimulation syndrome (OHSS) during pregnancy are both benign conditions where the ovaries are enlarged with presence of multiple thin-walled cysts. Differential diagnosis is ovarian malignancy. Hyperandrogenism with resultant maternal virilization could be seen in some cases of HL as well as in androgen secreting tumours. We report the case of a 41 years old lady underwent ovulation induction due to secondary infertility. She had recurrent hospital admission with abdominal pain and huge multicystic enlargement of both ovaries. She developed virilisation features by 35 weeks of pregnancy. Lower segment caesarean section was done at 36 weeks gestation for breech presentation with intra uterine growth restriction. Magnetic resonance imaging (MRI) confirmed benign nature of the cysts. Ovarian cysts and hyperandrogenism gradually resolved by three months of delivery. Awareness, judicious imaging and close monitoring in such cases can result in live birth and avoid oophorectomies.
 Keywords: Hyperandrogenism; Hirsuitism; Virilism; Polycstic ovary syndrome; Ovarian hyper stimulation; Ovulation induction; Hyperreactio luteinalis; Ovarian cysts.

Hyperreactio Luteinalis and an Incidentally Detected FIGO IIIB Borderline Tumor of the Ovary in Pregnancy - Diagnostic, Therapy and Review of Literature

Adnexal masses affect 2-10% of all pregnancies. The highest incidence of 1-6% can be seen in the first trimester, with a high rate of spontaneous remission. Two percent of these masses are malignant neoplasms or borderline tumors. A rare benign mass of the adnexa in pregnancy is hyperreactio luteinalis characterised by bilateral, multicystic ovaries with a frequent occurrence in the 3rd trimester. Clinical signs include maternal hyperandrogenaemia with virilisation, hyperemesis, nonspecific abdominal pain or laboratory findings of hyperthyroidism and elevated ß-HCG. Hyperreactio luteinalis does not require therapy due to complete spontaneous remission postpartum, but is often treated surgically in graviditate. In our case we report a first-time gravida in the 31st week of pregnancy with a symptomatic 25-cm multicystic, partly solid mass. After antenatal corticosteroid therapy, an exploratory laparotomy with right adnexectomy was performed on suspicion of malignancy. Histology revealed a hyperreactio luteinalis with an incidental finding of a serous borderline tumor of the ovary (FIGO IIIB). At 33 weeks of gestation, a pathological CTG was observed, and an urgent secondary caesarean section by re-longitudinal laparotomy was performed. Postpartum completion surgery revealed no further neoplastic cells.

Sonographic Comparison between PCOS, Hyperstimulated Ovaries and Hyperreactio Luteinalis

Background: Polycystic Ovarian Syndrome (PCOS) is a highly prevalent endocrinological disorder of females characterized by hirsutism, infertility and bilaterally enlarged multicystic ovaries.It is also a risk factor for development of Ovarian Hyperstimulation Syndrome (OHSS) and Hyperreatcio luteinalis (HL). Aim: To identify the value of ultrasonography in distinguishing these three interlinked pathologies and highlight the need for additional relevant investigations to make confirmatory diagnosis of any of them. Method: We conducted a systematic search by using Google Scholar and PubMed as search engines to retrieve all the available English-language literature on ultrasonographic findings of PCOS, OHSS and HL published from 2016 to 2021 while following PRISMA Statement. Results: Out of 316 articles retrieved ,only 9 articles were found that met the preset inclusion and exclusion criteria and of which there were 8 case reports and 1 narrative review. Conclusion: PCOS, OHSS and HL are interrelated ovarian morphological abnormalities with almost same ultrasonographic features.Correlation of these features with other relevant lab investigations and clinical parameters is therefore necessary to make an individual diagnosis out of them. Keywords: Ultrasound features, PCOS, Polycystic ovarian syndrome, Ovarian Hyperstimulation syndrome, Multicystic ovaries

HIPERREAÇÃO LUTEÍNICA EM GESTAÇÃO SIMPLES NORMAL: RELATO DE CASO

Hyperreactio luteinalis (HL) is a very rare and benign condition related to pregnancy, in which there is a massive and functional increase in ovaries, occupied by multiple benign theca lutein cysts. It generally does not affect pregnancy course and can be observed and diagnosed incidentally in third trimester. The cysts and, consequently, the ovaries regress spontaneously after delivery due to decrease in hCG. This report describes a case of HL in a 29-year-old primiparous woman with dyspnea, pelvic pain and clinical signs of hyperandrogenism, with the ovaries enlarged on transvaginal ultrasound. The conduct was expectant and, during delivery, a biopsy was performed, which confirmed the diagnosis. The knowledge of benign and self-limited disease defends a conservative treatment and helps in differentiation between HL and other malignant diseases. It is, therefore, essential that obstetricians are familiar with HL to guide the correct management and avoid unnecessary iatrogenic procedures.

A Case of Hyperreactio Luteinalis Complicated With Biochemical Hyperandrogenism, Symptomatic Hyperthyroidism and Preeclampsia

Background: Hyperreactio luteinalis (HL) describes the development of multiple large ovarian cysts during pregnancy, which regress post-partum. We report a case of HL complicated with preeclampsia, biochemical hyperandrogenism and hyperthyroidism. Clinical Case: A 31-year-old non-obese Chinese woman presented at 14-week gestation for lower abdominal pain. USG showed a single fetus, multiple ovarian cysts with largest measured 39.5ml. She complained of hand tremor, palpitation but no vomiting. She had no goiter, orbitopathy or family history of thyroid disease. fT4 was 23.1pmol/L (normal: 9.8-19.8pmol/L) and TSH was <0.01mIU/L. Anti-TG, anti-TPO and anti-TSHR antibodies were negative. She had history of silent miscarriage at 6-week gestation in her first pregnancy 2 years ago, USG showed normal ovaries at that time. Carbimazole was started at 16-week gestation for fT4 26.6pmol/L (normal: 9.4-18.5pmol/L). The largest ovarian cyst increased to 130ml at 19-week gestation. Serum β-hCG was 251926IU/L (normal: 4060-165400IU/L). HL with hCG-mediated hyperthyroidism was suspected. Serum total testosterone was 22.9nmol/L (normal: 2.2-10.7nmol/L) and serum androstenedione was 70.5nmol/L (normal: 0.28-9.81nmol/L). Ferriham Gallwey score was 4. fT4 fell to 13.8pmol/L (normal: 8.8-17.0pmol/L) but TSH remained suppressed. Carbimazole was stopped at 22-week gestation with no rebound in fT4 level. She developed preeclampsia and GDM at 27-week gestation. IUGR was evident despite decreasing β-hCG level and ovarian cyst shrinkage. She had emergency LSCS for severe preeclampsia at 33-week gestation. A 1510g female baby with normal genitalia was delivered. Placenta pathology was normal. 2 days after delivery, β-hCG fell to 7081IU/L; fT4 was 9.9pmol/L (normal: 9-19pmol/L) and TSH was 0.25mIU/L (normal: 0.35-4.5mIU/L). Clinical Lessons: 1) hCG stimulates growth of ovarian stroma and androgen secretion, results in virilization in 30% of HL patients. However, only 5% of patients had hyperthyroidism. LH and hCG are structurally similar and bind to the same receptor. In contrast, hCG is a weak agonist of TSH receptor: a hCG level of more than 100000IU/L is required to cause clinical thyrotoxicosis. Since 30% of HL patients have normal hCG level, this may explain the lower incidence of hyperthyroidism than hyperandrogenism. 2) Degree of maternal virilization does not correlate with testosterone level. Study by Condic et al. found significant overlap of testosterone levels in women with (13.7-197.5nmol/l) and without (6.2-37.3nmol/l) virilization. Genetic polymorphism of androgen receptor may account for the different clinical manifestation. Fetal virilization is rare, due to protective role of placental aromatase. 3) Elevated hCG in apparently “normal” singleton pregnancy may be due to poor placentation in early gestation and is a risk factor for preeclampsia and IUGR in HL patients.

Hyperreactio luteinalis incidentally found in the second trimester of pregnancy with normal hCG levels: a case report

Hyperreactio luteinalis (HL) is a rare entity in which both ovaries are multicystic and enlarged under the action of human chorionic gonadotropin (hCG), mostly seen in the third trimester of pregnancy. This benign condition is usually asymptomatic and doesn’t need any specific treatment, as the ovaries spontaneously reduce in size after birth. This is a case report of a 33-year-old woman diagnosed with hyperreactio luteinalis during the second trimester of her induced pregnancy. An ultrasound scan at 22 weeks of gestation revealed bilateral multicystic enlarged ovaries along with multiple fetal malformations and hydropsfetalis. Usually, HL is most commonly seen in situations in which there are high levels of hCG, but our patient had normal levels of hCG during all her pregnancy, which makes our case even rarer. In conclusion, the most important challenge when faced with HL is to differentiate between it and other differential diagnosis especially malignant tumors, because unlike them, this benign condition doesn’t need surgical treatment.

Hyperreactio Luteinalis (Multiple Luteinized Follicle Cysts): A Report of 10 Cases.

Hyperreactio luteinalis is a rare entity arising in pregnancy and in the setting of gestational trophoblastic diseases (ie choriocarcinoma, molar pregnancy) that presents with, typically, bilateral ovarian enlargement due to numerous follicle cysts. While the phenomenon is benign and spontaneously regresses following delivery or treatment, a specimen may be seen in pathology when oophorectomy or cystectomy is performed to exclude malignancy or to manage acute complications such as torsion. Such resections may exhibit overlapping microscopic features with cystic granulosa cell tumors. We thus reviewed 10 cases of hyperreactio luteinalis in the setting of pregnancy, the largest pathologic cohort to date, to highlight notable features of this disorder. Patients ranged from 22 to 30 yr old. Most patients (n=6) presented at time of cesarean section with incidentally discovered ovarian masses. Three patients presented in the postpartum period, and 1 underwent surgery at 28 wk gestation due to the finding of a unilateral ovarian mass. The ovaries ranged from 8.5 to 29 cm and were multicystic and bilateral in 8 of the cases. Histologic examination demonstrated multiple, variably sized cystic follicles lined by a granulosa cell layer of varying thickness and theca cells with marked eosinophilic cytoplasm. Stromal edema was often prominent, with theca cells occasionally noted in nests, cords, and as single cells in foci of edema. Mitoses were generally seen more often in the granulosa cell layer (mean=2.6 per high power fields) compared with the theca cell layer (mean=1 per 10 high power fields). This series documents the key features of hyperreactio luteinalis that differentiate it from the other benign mass forming lesions encountered in pregnancy, most notably large solitary follicle cyst of pregnancy and puerperium, as well as cystic granulosa cell tumors, especially the juvenile variant, which may also present during pregnancy. Of particular use in differentiating them from juvenile granulosa cell tumor is the absence of pale or vacuolated cytoplasm and solid growth of granulosa cells in cases of hyperreactio luteinalis.

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

BackgroundElevated 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.MethodsA 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.ResultsIn 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.ConclusionFrom 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.

Hiperreacción luteínica durante el tercer trimestre del embarazo. Reporte de caso

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.

Hemorrhagic infarction of hyperreactio luteinalis in spontaneously conceived singleton pregnancy: An uncommon cause of late third-trimester ovarian accident

Ovarian masses complicate about 1%–2% of all pregnancies while posing diagnostic and therapeutic challenges. Most are benign tumors but occasionally, functional cysts, such as theca lutein cysts, may be encountered in pregnancy. These are bilateral ovarian masses associated with high levels of β-human chorionic gonadotropin, thus seen mostly in the settings of trophoblastic diseases, multiple gestations, or following treatment for subfertility. It has also been reported in spontaneous singleton pregnancies. Clinical presentation is variable, ranging from asymptomatic conditions discovered incidentally to acute gynecological emergencies due to ovarian accidents. We report the case of a 29-year-old gravida 2, para 1 + 0, with estimated gestational age of 39 weeks plus 1 day, who had a previous emergency lower segment cesarean section (ELSCS) 3 years ago and presented with 1-h history of sudden-onset, sharp, right-sided, abdominal pain of increasing intensity. There were no history of trauma and no evidence of labor. On examination, she was a young gravid woman, at term, in severe painful distress. Baseline investigations were done, and she had ELSCS and exploratory laparotomy with a right salpingo-oophorectomy for twisted and hemorrhagic right adnexa discovered. Pathologic evaluation of the right ovary revealed an enlarged, dark brown, lobulated ovarian mass comprising multiple cortical cysts separated by a thin wall and containing dark brown fluid, with gray brown and hemorrhagic solid areas. A histologic diagnosis of hemorrhagic infarction on the background of multiple theca lutein cysts in pregnancy was made. She had a good postoperative recovery.

Diagnosis of hyperreactio luteinalis: A report of two cases

Diagnosis of hyperreactio luteinalis: A report of two cases

Continuous transabdominal drainage of ascites for management of hyperreactio luteinalis with massive ascites

Continuous transabdominal drainage of ascites for management of hyperreactio luteinalis with massive ascites

EP17.21: A case report of sacrococcygeal teratoma with bilateral theca lutein cysts

Sacrococcygeal teratoma (SCT) is the most common congenital germ cell tumour, with an incidence of 1 in 35,000-40,000 live births and a female predominance (3:1-4:1 ratio). Hyperreactio luteinalis (HL) is a rare entity in which there is bilateral benign functional multicystic ovarian enlargement during pregnancy which is most commonly seen in the third trimester and many times it is mistaken for ovarian malignancy and inadvertently operated upon. At a tertiary referral fetal medicine unit, Cairo University we reported a case presented at 32 weeks GA pregnant in a fetus with sacrococygeal teratoma type II according to American Academy of Pediatrics, Surgical Section (AAPSS). During the scan hyperreactio lutinalis was discovered accidentally by ultrasonography. Two days later the patient gave history of spontaneous rupture of membranes, premature labour and delivered by her physician by normal vaginal delivery. The fetus was delivered with respiratory distress, underwent NICU where the baby died 4 hours later. Three months later both ovaries resolved spontaneously and become completely normal diagnosed by ultrasonography. So we must not jump into diagnosis of ovarian malignancy in these cases. Please note: The publisher is not responsible for the content or functionality of any supporting information supplied by the authors. Any queries (other than missing content) should be directed to the corresponding author for the article.

EP30.08: Hyperreactio luteinalis: an often mistaken diagnosis

EP30.08: Hyperreactio luteinalis: an often mistaken diagnosis

Ovarian torsion of a term pregnancy – a new twist: review of the literature and management

Abstract Background Ovarian and adnexal torsions are gynecologic emergencies [Li Q, Li X, Zhang P. Ovarian torsion caused by hyperreactio luteinalis in the third trimester of pregnancy: a case report. Int J Clin Exp Med. 2015;8:19612–5; Ssi-Yan-Kai G, Rivain AL, Trichot C, Morcelet MC, Prevot S, Deffieux X. What every radiologist should know about adnexal torsion. Emerg Radiol. 2018;25:51–9; Huchon C, Fauconnier A. Adnexal torsion: a literature review. Eur J Obstet Gynecol Reprod Biol. 2010;150:8–12]. Case presentation Though rare in pregnancy, this case report describes a 32-year-old G0 at 39 + 3/7 weeks with a history of kidney stones and a simple ovarian cyst that presented to the labor floor with left lower quadrant pain. She underwent an induction of labor for persistent pain and had a subsequent primary section for a category II tracing. Conclusion During the section, a torsion x3 of the left infundibular ligament was noted. The pathology indicated an acute infarct – only one other case of a full-term torsion was found in the literature (Li Q, Li X, Zhang P. Ovarian torsion caused by hyperreactio luteinalis in the third trimester of pregnancy: a case report. Int J Clin Exp Med. 2015;8:19612–5).

Rapidly Progressive and Severe Hirsutism from Hyperreactio Luteinalis within a Background of Polycystic Ovary Syndrome

Gestational trophoblastic disease and hyperreactio luteinalis (HL) are rare, but important, etiologies of hyperandrogenism that need to be further studied. We present a case of rapidly progressing hirsutism and marked biochemical androgen excess in the context of pregnancy. A 26-year-old woman with a past medical history of obesity, prediabetes, and polycystic ovary syndrome presented with worsening hirsutism and markedly elevated testosterone levels. She was subsequently found to be pregnant, with extremely elevated levels of serum β-human chorionic gonadotropin. Subsequent work-up led to the identification of molar pregnancy and bilaterally enlarged ovaries, suggestive of HL. Following surgical intervention and therapy with methotrexate for invasive mole, she experienced improvement in both biochemical and clinical androgen excess features. With the prevalence of polycystic ovary syndrome, many women present to medical providers with hirsutism or other findings of hyperandrogenism. However, rapid progression of existing hirsutism or severe hirsutism should prompt more extensive evaluations to rule out rare etiologies. One such etiology found in pregnancy is HL, in which high levels of β-human chorionic gonadotropin can stimulate production of benign theca lutein cysts, leading to marked hyperandrogenism and virilizing symptoms.

Hyperreactio luteinalis: A clinical dilemma

Hyperreactio luteinalis (HL) is a pregnancy-associated benign condition with bilateral, rarely unilateral, ovarian enlargement caused by the presence of multiple cystic follicles secondary to β-human chorionic gonadotropin stimulation. Here, we report the case of a 24-year-old second gravida with a singleton pregnancy, with pregnancy-induced hypertension, who presented to the casualty with the chief complaints of headache and dizziness. The patient was later referred to the department of obstetrics and gynecology for further evaluation. On ultrasonography, a single live intrauterine pregnancy of 21-week gestation and bilateral enlarged cystic ovaries were revealed. With a clinical suspicion of ovarian tumor, bilateral oophorectomy was done, and the specimens were sent for histopathological study. On microscopy, the sections showed ovaries with numerous theca lutein cysts with hyperplasia and luteinization of the theca cells. Thus, surgical treatment is required for most of these patients. HL does not require any specific treatment except observation, unless complications occur.

EP16.02: Hyperreactio luteinalis: an often mistaken diagnosis

Hyperreactio luteinalis (HL) is a rare entity in which there is bilateral benign functional multicystic ovarian enlargement during pregnancy which is most commonly seen in the third trimester and many times it is mistaken for ovarian malignancy and inadvertently operated upon. Sacrococcygeal teratoma (SCT) is the most common congenital germ cell tumour, with an incidence of 1 in 35,000-40,000 live births and a female predominance (3:1-4:1 ratio). At a tertiary referral fetal medicine unit, Cairo University we reported a case presented at 32 weeks GA pregnant in a fetus with sacrococygeal teratoma. During the scan hyperreactiolutinalis was discovered accidentally by ultrasonography. Two days later the patient gave history of spontaneous rupture of membranes, premature labour and delivered by her physician by normal vaginal delivery. The fetus was delivered with respiratory distress, underwent NICU where the baby died 4 hours later. Three months later both ovaries resolved spontaneously and become completely normal diagnosed by ultrasonography. So we must not jump into diagnosis of ovarian malignancy in these cases. Supporting information can be found in the online version of this abstract Please note: The publisher is not responsible for the content or functionality of any supporting information supplied by the authors. Any queries (other than missing content) should be directed to the corresponding author for the article.

Diagnostic Imaging of Pregnant Women - The Role of Magnetic Resonance Imaging.

SummaryBackgroundPresentation of magnetic resonance imaging (MRI) findings in pregnant women in the Department of Diagnostic Imaging, Institute of Mother and Child, Warsaw, Poland.Material/MethodsForty-three symptomatic pregnant women underwent MRI between 9 and 33 weeks of gestation (mean of 23 weeks). Moreover, we included 2 pregnant women who underwent fetal MRI and had incidental abnormalities.ResultsIn 9 cases, we excluded the suspected brain abnormalities. In 4 cases, we found unremarkable changes in the brain without clinical significance. One patient was diagnosed with multiple sclerosis, one with cortical dysplasia, one with pineal hemorrhage and one with a brain tumor. On abdominal MRI, 2 patients had normal findings, one patient had colon cancer with a hepatic metastasis, one patient had a hepatic angioma, one patient had an extraadrenal pheochromocytoma, one patient had an abscess in the iliopsoas muscle, 9 patients had myomas, two patients had ovarian simple cysts, two endometrial cysts, three dermoid cysts, one patient had sacrococcygeal teratoma, one patient had a cystadenofibroma (partial borderline tumor), one patient had an androgenic ovarian tumor and two patients had hyperreactio luteinalis. One patient was diagnosed with transient osteoporosis of the hip and one with a stress fracture of the sacral bone.ConclusionsMagnetic resonance imaging is the best imaging modality for pregnant women. Although ultrasonography is the method of choice, doubtful cases as well as structures that cannot be examined with ultrasonography can be non-invasively evaluated with MRI.