Solitary luteinized follicle cyst is a rare cause of ovarian enlargement during pregnancy and puerperium. Only rare cases of this clinical condition have been reported in the literature. We report a case of a large solitary luteinized follicle cyst arising in a 32-year-old multipara four days post partum which required postpartum surgical intervention. The perinatal outcome is usually good, with liveborn infants being delivered at or near term. The complications of the disease include ovarian torsion, intra-cystic haemorrhage, and rupture. The condition should be included in the differential diagnostic workup whenever a growing simple cyst is encountered during pregnancy and puerperium.Key words: luteinized follicle cyst; pregnancy; puerperium

Ali, Ali M

Hummeida, Moawia E

Mostafa, El Rayah M

Satir, Ali Abdel

English

AJOL - African Journals Online

 © Sudan JMS Vol. 10, No.4. Dec 2015            169  Vtáx exÑÉÜà  Large solitary luteinized follicle cyst of pregnancy and puerperium Moawia E Hummeida1, Ali Abdel Satir2, Ali M Ali3, El Rayah M. Mostafa4  ABSTRACT Solitary luteinized follicle cyst is a rare cause of ovarian enlargement during pregnancy and puerperium. Only rare cases of this clinical condition have been reported in the literature. We report a case of a large solitary luteinized follicle cyst arising in a 32-year-old multipara four days post partum which required postpartum surgical intervention. The perinatal outcome is usually good, with liveborn infants being delivered at or near term. The complications of the disease include ovarian torsion, intra-cystic haemorrhage, and rupture. The condition should be included in the differential diagnostic workup whenever a growing simple cyst is encountered during pregnancy and puerperium. Key words: luteinized follicle cyst; pregnancy; puerperium.  varian masses are relatively common in pregnancy, with a prevalence of 2.3–5.4% being reported in prospective studies owing to the increase use of ultrasound examination in pregnancy. Functional cysts represent the vast majority of these masses, followed by benign cystic teratomas, serous cystadenomas, paraovarian cysts, mucinous cystadenomas, endometriomas and rarely malignant tumors1. The majority of adnexal cysts are small (<5 cm), remain asymptomatic and tend to resolve before birth2, 3, mostly during the first trimester4. Simple cysts of > 5 cm in diameter are much less common (representing about 4% of the total). They still tend to resolve spontaneously; however, compared with smaller cysts, they are more likely to develop torsion or haemorrhage, require surgical removal during pregnancy or even be associated with     malignancy,      especially      when ______________________________________________ 1. School of Medicine, Alneelain University, Sudan 2. Consultant Histopathologist, Khartoum Sudan 3. Alneelain university 4. Radiology Department, Suba University hospital, Sudan  *Correspondence to: Moawia Hummeida. Email:abuawa25@gmail.com Phone: 00249 912332067 growing3,5,6. A rapidly enlarging ovarian mass in pregnancy poses significant diagnostic problems.  Large luteinized cysts of pregnancy are an uncommon type of cystic mass particular to pregnancy, characterized by the combination of a benign appearance and a tendency to enlarge rapidly, eventually becoming symptomatic and most often necessitating surgery. PRESENTATION OF CASE:  Our patient was a 32-years-old housewife G5 P4. All deliveries were normal at home, she has two living boys and two girls, and youngest is three years old.  She presented to the clinic with a history of vague abdominal pain, gradually increasing discomfort and distension four days following spontaneous full term vaginal delivery at home. Her medical and family history was non-significant. Her previous obstetric history was normal. She has no regular antenatal care during current pregnancy; she has been seen once by a midwife and has no history of laboratory or ultrasound examination. Pregnancy and delivery were reported as uneventful. She gave birth to a healthy 3.700 g male infantat home four days before admission and she is breast OHummeida et. al                Large solitary luteinized follicle cyst of pregnancy and puerperium © Sudan JMS Vol. 10, No.4. Dec 2015                    170 feeding. On admission her general condition was good. Her abdomen was grossly distended, soft with mild tenderness and no rebound pain. The remainder of the examination was normal apart from lower limb oedema. Ultrasound examination revealed a large solitary, unilocular cyst with a thin, smooth wall and homogeneous anechoic content. The cyst has no septae, lacked mural blood flow and was not associated with ascites. It was located in the central abdominal area above and anterior to the postpartum uterus CT scan report showed a huge pelvi-abdominal cystic structure, arising from the left side of the pelvis and extending upward and lies just below the liver.  It displaces the bowel loops aside. It has thin walls, no enhancing septations and not associated calcifications or soft tissue component. The cyst measures 27X16 cm. Uterus is bulky with normal walls with normal urinary bladder. No hydronephrosis or hydroureters. No ascites or enlarged lymph nodes. Other laboratory tests were normal and CA125 was 5.5 m/u. Laparotomy was carried out and a right side huge solitary unilocular cyst measuring -26 x 25 x 40 cm was identified (Figure 1). The left ovary was normal; there were no septa, no accompanying ascites, and no other abdominal or pelvic abnormalities were detected. A right Salpingo-oopherectomy was performed. Her postoperative course was uneventful and was discharged home in good condition. Macroscopic examination shows 6,200 g left ovarian cyst measuring40 cm in maximum diameter. Cut surface shows unilocular cyst containing turbid straw coloured fluid.  Microscopy shows ovarian cyst lined by luteinized granulosacells (Figure 2). No evidence of neoplasm of adequate sample examined. The features are those of large solitary luteinized follicle cyst of pregnancy and puerperium. DISCUSSION:  In spite of a very large cyst (27X16 cm) (6,200 g) which almost fills the entire abdominal cavity, the patient remained asymptomatic during pregnancy, only to be diagnosed in early puerperium by ultrasound due to persistent distension and discomfort. The pathogenesis of large luteinized cysts of pregnancy is unclear. Itsoccurrence in pregnancy suggests stimulation with Human chorionic gonadotropin (HCG)7, or increased tissue sensitivity to hCG7. However, these cysts can also appear up to 3 months postpartum8,9, when HCG has normally become undetectable. Thus HCG may not be the only contributing factor. In such cases, Clement and Scully postulated that the development of these cysts is initially (prenatally) stimulated by HCG, albeit to a size that may remain unnoticed. A rise in the pituitary gonadotropins (follicle-stimulating hormone/luteinizing hormone), if there is no lactation postpartum, may be responsible for their continuing enlargement7. The ultrasound appearance of large luteinized cysts of pregnancy facilitates their differential diagnosis from other, more common, adnexal masses. Follicular and corpus luteum cysts are relatively common, but they rarely exceed 6–8 cm in diameter10,11. Pregnancy luteoma and hyperreactioluteinalis only occur during pregnancy. Both of these masses can cause maternal virilization (25–35%), with luteomas also causing virilization of the female fetus in about one-third of cases7,12. When diagnosed prenatally, they appear as complex ovarian masses. Luteomas are typically multiple and often occur bilaterally. Their appearance is predominantly solid, but they may also have cystic areas and are typically 6–10cmHummeida et. al                Large solitary luteinized follicle cyst of pregnancy and puerperium © Sudan JMS Vol. 10, No.4. Dec 2015                    171  Figure 1: Left Large solitary luteinized follicle cyst of pregnancy and puerperium. in size, occasionally reaching 20 cm10,13. Hyperreactioluteinalis is typically bilateral and is characterized by ovarian enlargement caused by numerous luteinized follicular cysts, often with hemorrhagic areas. In contrast to luteoma, hyperreactioluteinalis is seen in conditions with abnormally high levels of HCG (e.g. twins,  hydatidiform  mole,   hydrops   and    Figure 2: Microscopy shows ovarian cyst lined by luteinized granulosa cells.choriocarcinoma)10,12. A diagnostic problem may arise with serous cystadenomas, which are usually unilocular (occasionally bilocular), with a thin, smooth wall, a thin, regular septum (when present) and homogeneous anechoic content14. The same is true for benign cystic teratomas, which can vary from entirely cystic to echogenic, with different degrees of density and shadowing15. Such cysts do not usually demonstrate the rapid growth rate reported for large luteinized cysts of pregnancy, and only rarely require removal during pregnancy because of complication16. The potential for malignancy is always a concern when considering a rapidly enlarging ovarian mass. Fortunately, less than 1% of ovarian masses in pregnancy are malignant, about one-third of them being germ cell tumors17, and therefore the positive prognostic value of any ultrasound sign is low. Still, close monitoring would be recommended for large masses, especially as the reliability of ancillary tests (e.g. Doppler indices, CA 125, -HCG or alpha-fetoprotein) is reduced as a result of the physiological adaptations of pregnancy. As with any adnexal mass, the management options include either expectant follow-up or surgical removal. Data from series in pregnancy indicate that the risk for torsion in cysts 6–8 cm in diameter is 22%6; and, one in nine cysts of more than 5 cm will require surgery and this rate increases to 45% for symptomatic cysts19. Although published data specifically concerning large luteinized cysts of pregnancy are limited, their tendency to grow means that they are associated with higher risk, and removal was eventually required, either before delivery10,19 or during puerperium (as in our case)20,21. Fortunately, it appears that adnexal surgery in pregnancy is relatively safe.  Hummeida et. al                Large solitary luteinized follicle cyst of pregnancy and puerperium © Sudan JMS Vol. 10, No.4. Dec 2015                    172 CONCLUSION:  In conclusion, large luteinized follicular cysts of pregnancy are an uncommon type of cystic mass particular to pregnancy, which should be included in the differential diagnostic workup whenever a growing simple cyst is encountered. These cysts are characterized by the combination of a benign appearance and a tendency to enlarge rapidly, eventually becoming symptomatic and most often necessitating surgery. REFERENCES: 1. Bignardi T, Condous G. The management of ovarian pathology in pregnancy. Best Pract Res ClinObstet Gynaecol2009; 23: 539–548. 2. Condous G, Khalid A, Okaro E, Bourne T. Should we be examining the ovaries in pregnancy? Prevalence and natural history of adnexal pathology detected at first-trimester sonography. Ultrasound Obstet Gynecol2004; 24: 62–66. 3. Bernhard LM, Klebba PK, Gray DL, Mutch DG. Predictors of persistence of adnexal masses in pregnancy. Obstet Gynecol1999; 93:585–589. 4. Glanc P, Brofman N, Salem S, Kornecki A, Abrams J, Farine D. The prevalence of incidental simple ovarian cysts > or = 3 cm detected by transvaginal sonography in early pregnancy. J ObstetGynaecol Can2007; 29: 502–506. 5. Zanetta G, Mariani E, Lissoni A, Ceruti P, Trio D, Strobelt N, Mariani S. A prospective study of the role of ultrasound in the management of adnexal masses in pregnancy. BJOG2003; 110: 578–583. 6. Yen CF, Lin SL, Murk W, Wang CJ, Lee CL, Soong YK, Arici A. Risk analysis of torsion and malignancy for adnexal masses during pregnancy. Fertil Steril2009; 91: 1895–1902. 7. Clement PB, Scully RE. Large solitary luteinized follicle cyst of pregnancy and puerperium: a clinicopathological analysis of eight cases. Am J Surg Pathol1980; 4: 431–438. 8. homas S, Krishnaswami H, Seshadri L. Large solitary luteinized follicle cyst of pregnancy and puerperium. 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Buy JN, Ghossain MA, Sciot C, Bazot M, Guinet C, Prévot S, Hugol D, Laromiguiere M, Truc JB, Poitout P. Epithelial tumors of the ovary: CT findings and correlation with US. Radiology1991; 178: 811–818. 15. Caspi B, Levi R, Appelman Z, Rabinerson D, Goldman G, Hagay Z. Conservative management of ovarian cystic teratoma during pregnancy and labor. Am J Obstet Gynecol2000; 182: 503–505. 16. .Mavromatidis G., Sotiriadis A., Dinas K., Mamopoulos A. and Rousso D. Large luteinized follicular cyst of pregnancy. Ultrasound ObstetGynecol 2010; 36: 517–520.  17. Leiserowitz GS, Xing G, Cress R, Brahmbhatt B, Dalrymple JL, Smith LH. Adnexal masses in pregnancy: how often are they malignant?Gynecol Oncol2006; 101: 315–321. 22  18. .Lomme M, Kostadinov S, Zhang C.Large solitary luteinized follicle cyst of pregnancy and puerperium: report of two cases. DiagnPathol. 2011 Jan 10; 6:3. Epub 2011 Jan 10 19. Fang YM, Gomes J, Lysikiewicz A, Maulik D. Massive luteinized follicular cyst of pregnancy. 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Large solitary luteinized follicle cyst of pregnancy and puerperium

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Large solitary luteinized follicle cyst of pregnancy and puerperium

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