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Cover page of the Journal of Health Sciences


 
 Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 12  |  Issue : 3  |  Page : 248-250

Hyperreactio luteinalis: A clinical dilemma


Department of Pathology, Bharati Vidyapeeth (Deemed to be) University Medical College and Hospital, Sangli, Maharashtra, India

Date of Web Publication15-Oct-2019

Correspondence Address:
Dr. Seema R Balasubramaniam
C5, Bharati Girls' Hostel-3, Bharati Vidyapeeth Deemed University Medical College and Hospital, Sangli - 416 414, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/kleuhsj.kleuhsj_182_18

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  Abstract 


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.

Keywords: Clinical dilemma, hyperreactio luteinalis, pregnancy-induced hypertension


How to cite this article:
Balasubramaniam SR, Pawar VR. Hyperreactio luteinalis: A clinical dilemma. Indian J Health Sci Biomed Res 2019;12:248-50

How to cite this URL:
Balasubramaniam SR, Pawar VR. Hyperreactio luteinalis: A clinical dilemma. Indian J Health Sci Biomed Res [serial online] 2019 [cited 2019 Nov 19];12:248-50. Available from: http://www.ijournalhs.org/text.asp?2019/12/3/248/269198




  Introduction Top


Hyperreactio luteinalis (HL) is a pregnancy-associated benign condition with bilateral, rarely unilateral, ovarian enlargement caused by the presence of multiple luteinized cystic follicles secondary to β-human chorionic gonadotropin (HCG) stimulation.[1] HL is commonly associated with gestational trophoblastic diseases (GTDs), multiple pregnancies, use of ovulatory induction drugs, and fetal hydrops.[2] The cause of this is unknown, but it is believed to be related to elevated levels of, or abnormal ovarian response to, β-HCG and pituitary gonadotropins. The ovaries in HL are symmetrical with uniform-sized theca lutein cysts. This benign condition may imitate enlarged ovaries in ovarian hyperstimulation syndrome (OHSS) or ovarian malignancy. OHSS is a consequence of iatrogenic ovarian induction, but in rare instances, it can occur spontaneously, particularly in hypothyroidism, pituitary gonadotropin-secreting adenoma, pregnant women with polycystic ovarian syndrome, or mutation in follicle-stimulating hormone or luteinizing hormone receptors.


  Case Report Top


Here, we report the case of a 24-year-old woman, a second gravida with a singleton pregnancy, a known case of pregnancy-induced hypertension, who was referred to the department of obstetrics and gynecology, with the complaints of headache and dizziness. Informed consent was taken in accordance to the ethical committee of the institute, prior to the case reporting. On admission, her vitals were stable, blood pressure was recorded as 160/100 mmHg, and heart rate was 90 beats/min. The blood pressure was monitored regularly at 15-min interval. Laboratory data showed a normal liver, renal function tests with normal coagulation parameters, an hemoglobin level of 11 g/dl, and a platelet count of 250,000 μL. Transabdominal ultrasonography revealed a single live intra-uterine pregnancy of 21-week gestation and bilateral enlarged cystic ovaries. The patient was counseled for repeat cesarean section, and consent was taken after explaining the risks and the benefits of the procedure.

The patient was then posted for emergency lower-segment cesarean sectioning. During the procedure, bilaterally enlarged grape-like cystic ovaries were noted. With a clinical suspicion of ovarian tumor, bilateral oophorectomy was done, and the specimens were sent for histopathological study.

The pregnancy was allowed to continue; however, we lost in touch with the patient, and hence, follow-up could not be done about the pregnancy and the outcome.

Gross

Bilateral enlarged multiple ovaries were received. The left ovary weighed 450 g and measured 9 cm × 5 cm × 4.5 cm. The right ovary weighed 470 g and measured 11 cm × 6 cm × 6 cm, with attached fallopian tube measuring 4 cm in length. The external surface was dark brown. Cut section showed multiple, thin-walled cysts filled with hemorrhagic fluid [Figure 1].
Figure 1: Multiple, thin-walled cysts with hemorrhagic fluid

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Microscopy

Sections showed an ovary with numerous theca lutein cysts with hyperplasia and luteinization of the theca cells. The stroma was edematous and showed extensive focal luteinization [Figure 2] and [Figure 3].
Figure 2: Follicular cyst wall lined by luteinized cells

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Figure 3: Luteinized stromal cells present within the edematous stroma

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  Discussion Top


HL is associated with conditions such as hydatidiform mole, choriocarcinoma, fetal hydrops, and multiple gestations.[1] It is most often bilateral and found incidentally at the time of cesarean section. However, HL may present during any trimester as an abdominal mass or acute abdomen. The natural course is postpartum regression.[3] It is estimated that approximately 60% of the cases of HL is not associated with trophoblastic disease, and these cases present with a normal singleton pregnancy.[1],[3] Depending on the size of the masses, patients are either asymptomatic or present with pain due to increased intra-abdominal pressure, torsion, or intracystic hemorrhage. This may necessitate oophorectomy; however, most of the time, it is undertaken due to a misdiagnosis of ovarian tumors such as mucin-secreting cystadenomas or carcinomas. Virilization due to hyperandrogenism can occur in as many as 25% of the affected patients.[4] The frequency of HL in women with GTD ranges from 10% (by clinical examination) to 40% (by ultrasonography).[5] Prolactin and estradiol are suspected to have a role in the pathogenesis or maintenance of HL.[1] OHSS is also characterized by enlarged bilateral multicystic ovaries, occurring in the first trimester, and sometimes termination of the pregnancy is unavoidable. OHSS is a severe complication in pregnancy following ovulation induction with exogenous gonadotropins.[6] The pathophysiology of HL cysts is similar to OHSS; however, they can be differentiated by the fact that OHSS is iatrogenic, whereas HL is a spontaneous occurrence.[7] It is essential to exclude these through differential diagnosis via a wedge biopsy and frozen section to avoid unnecessary surgical excision.[3]

HL may mimic ovarian malignancies such as mucinous borderline tumor of intestinal type; however, they are differentiated by the fact that they have smaller thin-walled cysts and not as much solid component as seen in HL. They can also be differentiated from the same, based on β-HCG levels, ovarian tumor markers, and imaging modality correlation such as ultrasonography and magnetic resonance imaging.[7] On ultrasound examination, HL is characterized by a large adnexal mass that consists of many thin-walled small theca lutein cysts, giving it the appearance of a “spoke wheel.”[2],[4] HL may also explain the underlying cause of preeclampsia because symptoms such as hyperemesis, eclampsia, and preterm contractions may be associated with high β-HCG levels and may be associated with incomplete placental invasion and incomplete angiogenesis.[3] Regression of HL occurs during puerperium, and in cases associated with GTD, regression occurs 2–12 weeks after uterus evacuation.[1],[5] In addition, Cho et al. had reported complete resolution of the condition at the 7th week after vaginal delivery.[8]

Surgical treatment is required for most of these patients. First, cesarean section is preferred because of obstruction of the birth canal by enlarged ovaries. Second, we can take a biopsy of the ovarian cyst or wedge resection, and even sometimes, oophorectomy because of ovarian torsion, infarction, and hemorrhage or to rule out ovarian malignancy. But HL does not need any specific treatment except observation, unless complications occur.[6],[7]


  Conclusion Top


Awareness of HL among pathologists, radiologists, and gynecologists will help in avoiding unnecessary oophorectomies.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Kurman RJ. Blaustein's Pathology of the Female Genital Tract. 6th ed. Baltimore, USA: Springer Publishing; 2004. p. 690-1.  Back to cited text no. 1
    
2.
Abbas AM, Talaat E, Gamal E, Michael A, Hanna S, Ali MN. An unusual case of asymptomatic hyperreactio luteinalis present at cesarean section of a spontaneous singleton pregnancy. Middle East Fertil Soc J 2017;30:160-2.  Back to cited text no. 2
    
3.
Atis A, Cifci F, Aydin Y, Ozdemir G, Goker N. Hyperreactio luteinalis with preeclampsia. J Emerg Trauma Shock 2010;3:298.  Back to cited text no. 3
[PUBMED]  [Full text]  
4.
Van Holsbeke C, Amant F, Veldman J, De Boodt A, Moerman P, Timmerman D. Hyperreactio luteinalis in a spontaneously conceived singleton pregnancy. Ultrasound Obstet Gynecol 2009;33:371-3.  Back to cited text no. 4
    
5.
Clement PB, Young RH. Atlas of Gynecologic Surgical Pathology. 2nd ed. USA: Saunders Elsevier; 2008. p. 291-2.  Back to cited text no. 5
    
6.
Yang C, Wang H, Zou Y, Cui W, Yang J, Li T, et al. Hyperreactio luteinalis after delivery: A case report and literature review. Int J Clin Exp Med 2015;8:6346-8.  Back to cited text no. 6
    
7.
Skandhan AK, Ravi V. Hyperreactio luteinalis: An often mistaken diagnosis. Indian J Radiol Imaging 2014;24:84-6.  Back to cited text no. 7
[PUBMED]  [Full text]  
8.
Cho AR, Song TB, Kim HJ, Kim YH, Kim JW. Vaginal delivery in a spontaneously conceived singleton pregnancy complicated with hyperreactio luteinalis: A case report. J Womens Med 2011;4:53-6.  Back to cited text no. 8
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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