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Hyperreactio Luteinalis in a Normal Singleton Pregnancy: a Case Report

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Brazilian Journal of Development 50897 ISSN: 2525-8761 Hyperreactio luteinalis in a normal singleton pregnancy: a case report Hiperreação luteínica em gestação simples normal: relato de caso DOI:10.34117/bjdv7n5-472 Recebimento dos originais: 07/04/2021 Aceitação para publicação: 03/05/2021 João Eduardo Andrade Tavares de Aguiar Discente do Departamento de Medicina da Universidade Federal de Sergipe – UFS Instituição: Universidade Federal de Sergipe – UFS Endereço: Rua Cláudio Batista, s/n – Santo Antônio, Aracaju–SE, 49060-100 E-mail: [email protected] Alessandra Aleixo Albuquerque Ginecologista e Obstetra pelo Hospital Universitário da Universidade Federal de Sergipe – HU/UFS Instituição: Universidade Federal de Sergipe – UFS Endereço: Rua Cláudio Batista, s/n – Santo Antônio, Aracaju–SE, 49060-100 E-mail: [email protected] Marcos Antônio Lima Carvalho Discente do Departamento de Medicina da Universidade Federal de Sergipe – UFS Instituição: Universidade Federal de Sergipe – UFS Endereço: Rua Cláudio Batista, s/n – Santo Antônio, Aracaju–SE, 49060-100 E-mail: [email protected] Artime Alves Costa Ginecologista e Obstetra no Hospital da Polícia Militar de Sergipe Médico pela Universidade Federal de Sergipe – UFS Instituição: Universidade Federal de Sergipe – UFS Endereço: Rua Cláudio Batista, s/n – Santo Antônio, Aracaju–SE, 49060-100 E-mail: [email protected] Bárbara Rhayane Santos Discente do Departamento de Medicina da Universidade Federal de Sergipe – UFS Instituição: Universidade Federal de Sergipe – UFS Endereço: Rua Cláudio Batista, s/n – Santo Antônio, Aracaju–SE, 49060-100 E-mail: [email protected] Marcella Andrade Tavares de Aguiar Discente de Medicina do Centro Universitário Tiradentes – UNIT/AL Instituição: Centro Universitário Tiradentes – UNIT/AL Endereço: Av. Comendador Gustavo Paiva, 5017 – Cruz das Almas, Maceió–AL, 57038-000 E-mail: [email protected] Mariana Cunha de Sousa Discente do Departamento de Medicina da Universidade Federal de Sergipe – UFS Instituição: Universidade Federal de Sergipe – UFS Brazilian Journal of Development, Curitiba, v.7, n.5, p. 50897-50907 may 2021 43 may. 2021 Brazilian Journal of Development 50898 ISSN: 2525-8761 Endereço: Rua Cláudio Batista, s/n – Santo Antônio, Aracaju–SE, 49060-100 E-mail: [email protected] Júlia Maria Gonçalves Dias Docente do Departamento de Medicina da Universidade Federal de Sergipe – UFS Doutora em Ciências da Saúde pela Universidade Federal de Sergipe – UFS Universidade Federal de Sergipe – UFS Endereço: Rua Cláudio Batista, s/n – Santo Antônio, Aracaju–SE, 49060-100 E-mail: [email protected] ABSTRACT 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. Keywords: Ovarian diseases. Ovarian Cysts. Theca cells. RESUMO A hiperreação luteínica (HL) é uma condição muito rara e benigna relacionada à gravidez, na qual há um aumento maciço e funcional dos ovários, ocupados por múltiplos cistos teca luteínicos benignos. Geralmente não afeta o curso da gravidez e pode ser observada e diagnosticada incidentalmente no terceiro trimestre. Os cistos e, consequentemente, os ovários regridem espontaneamente após o parto por conta da diminuição de hCG. Este relato descreve um caso de HL em primigesta de 29 anos com dispneia, dor pélvica e sinais clínicos de hiperandrogenismo, apresentando os ovários aumentados na ultrassonografia transvaginal. A conduta foi expectante e, durante o parto, foi realizada biópsia, que confirmou o diagnóstico. O conhecimento da doença benigna e autolimitada defende um tratamento conservador e auxilia na diferenciação entre a síndrome e outras doenças malignas. É, portanto, essencial que obstetras estejam familiarizados com a HL para direcionar o correto manejo e evitar desnecessários procedimentos iatrogênicos. Palavras-chaves: Doenças ovarianas. Cistos Ovarianos. Células tecais. 1 INTRODUCTION Hyperreactio luteinalis (HL) is a very rare and benign condition related to pregnancy, in which there is a massive, functional and bilateral enlargement of ovaries, Brazilian Journal of Development, Curitiba, v.7, n.5, p. 50897-50907 may 2021 43 may. 2021 Brazilian Journal of Development 50899 ISSN: 2525-8761 occupied by multiple benign theca lutein cysts1-3. About 100 cases of HL have been reported in literature, with the majority of cases in multiparous patients, with multiple pregnancies, with gestational trophoblastic disease or undergoing infertility treatment1,4. This condition is very rare in a single spontaneous pregnancy5-6. HL is a gestational syndrome that generally does not affect pregnancy course and can be observed and diagnosed incidentally in third trimester2,7. In most cases, there are no symptoms, but it can lead to nausea and vomiting, lower abdominal pain, loss of appetite, electrolyte disorders, ascites and signs of virilization (such as hirsutism and clitoromegaly)2. Although etiology is still unknown, it is believed the cysts' formation is associated with high serum level of human chorionic gonadotropin (hCG) or with increased ovarian sensitivity to hCG, causing increased production of ovarian androgens1. In this study, we report a primigravid patient with spontaneous bilateral ovarian enlargement during a normal singleton pregnancy, in which the diagnosis of HL was made in an obstetrics service in Sergipe. 2 CASE DESCRIPTION A 29-year-old primiparous patient, with no comorbidities, clinical, gynecological or surgical history, started normal prenatal single gestation with 10 weeks in a private service in Aracaju, Sergipe. The patient reported symptoms of dyspnoea and pelvic pain and she had clinical signs of hyperandrogenism, such as hirsutism, acne and alopecia. On transvaginal ultrasound (USG TVS), at 24 weeks, a solid cystic mass was observed in left ovary, with 89 cm3 (Figure 1), and an MRI was requested, which confirmed the presence of a multi- sept cystic mass. β-hCG was requested, resulting in 1084.13 mIU/mL. The conduct, after a literary review, was expectant, waiting for the moment of delivery to perform a biopsy. Brazilian Journal of Development, Curitiba, v.7, n.5, p. 50897-50907 may 2021 43 may. 2021 Brazilian Journal of Development 50900 ISSN: 2525-8761 Figure 1. Transvaginal ultrasound of left ovary, with 89 cm3, showing a multi-sept cystic mass in a pregnant woman at 24 weeks of gestation In obstetric ultrasonography with doppler, at 37 weeks, the mass reached 366 cm3, with numerous regular, thick and vascularized septa, in addition to showing another cyst in right ovary, with the same characteristics of the left, of 430 cm3. Previously, two other obstetric ultrasounds, performed at 33 and 34 weeks, demonstrated ovaries with the same characteristics. The 33-week ultrasound presented a left ovary with 247cm3 (Figure 2) and a right ovary with 262cm3 (Figure 3), and the 34-week ultrasound showed a left ovary with 284cm3 and a right ovary 299cm3. Figure 2. Transvaginal ultrasound of left ovary, 262 cm3, with multi-sept cystic mass in a pregnant woman at 33 weeks of gestation. Brazilian Journal of Development, Curitiba, v.7, n.5, p. 50897-50907 may 2021 43 may. 2021 Brazilian Journal of Development 50901 ISSN: 2525-8761 Figure 3. Transvaginal ultrasound of right ovary, 247 cm3, with multi-sept cystic mass in a pregnant woman at 33 weeks of gestation. There was no anticipation of delivery, and a cesarean section was performed at 40 weeks of gestation, associated with oophoroplasty for biopsy (Figures 4 e 5), in August 2017. The result showed in both ovaries luteinized follicular cysts, with hemorrhagic corpus lutein on the left. In September 2017, USG TVS was performed, which showed a reduction in volumes to 211cm3 and 247cm3 in right and left ovaries, respectively, followed by monitoring and USG TVS. In March 2019, another USG TVS showed ovaries with volumes 21.7cm3 to the right and 21.5cm3 to the left. Brazilian Journal of Development, Curitiba, v.7, n.5, p. 50897-50907 may 2021 43 may. 2021 Brazilian Journal of Development 50902 ISSN: 2525-8761 Figure 4. Left ovary during cesarean delivery. Figure 5. Oophoroplastia with wedge resection of left ovary during cesarean delivery. 3 DISCUSSION HL, also called theca lutein cyst, is a self-limited syndrome, with bilateral enlargement of ovaries, and is associated with theca lutein cysts and increased hCG levels, which is secreted by the trophoblast in first trimester, with a peak in the 9th week Brazilian Journal of Development, Curitiba, v.7, n.5, p. 50897-50907 may 2021 43 may. 2021 Brazilian Journal of Development 50903 ISSN: 2525-8761 of gestation (highest average level of 100,000
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