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CASE REPORT
Year : 2014  |  Volume : 3  |  Issue : 4  |  Page : 267-269

Maternal luteoma of pregnancy: A rare case report


Department of Pathology, Konaseema Institute of Medical Sciences and Research Foundation, Amalapuram, Andhra Pradesh, India

Date of Web Publication10-Dec-2014

Correspondence Address:
D Siva Ranjan
H. No. C-108, Phase- 2, Hill Colony, Vanasthalipuram, Hyderabad - 500 070, Andhra Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2277-8632.146639

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  Abstract 

Pregnancy luteoma is a rare non-neoplastic tumor like mass of the ovary. They are usually asymptomatic and found incidentally during imaging or surgery. They regress spontaneously after delivery. We present a case in a 28-year-old female patient with an ovarian mass, which was found incidentally during cesarean section. Histopathological examination showed features of luteoma of pregnancy. Recognition of this entity is important so that unnecessary oophorectomy, with concomitant risk to both the mother and the fetus, is avoided.

Keywords: Cesarean section, oophorectomy, ovary, pregnancy luteoma


How to cite this article:
Kumar R V, Ranjan D S, Rao B, Rao ES. Maternal luteoma of pregnancy: A rare case report. J NTR Univ Health Sci 2014;3:267-9

How to cite this URL:
Kumar R V, Ranjan D S, Rao B, Rao ES. Maternal luteoma of pregnancy: A rare case report. J NTR Univ Health Sci [serial online] 2014 [cited 2019 Jan 18];3:267-9. Available from: http://www.jdrntruhs.org/text.asp?2014/3/4/267/146639


  Introduction Top


Pregnancy luteoma was first described by Sternberg and Barclay in 1966. [1] Until, fewer than 200 cases of pregnancy luteoma have been reported. [2] In general, luteomas are asymptomatic and are found incidentally at the time of cesarean section or postpartum tubal ligation. [3] Luteoma of pregnancy is a benign, hyperplastic tumor like lesion of the ovary. Hyper secretion of androgens occurs in approximately 25% of women with pregnancy luteoma; from 10% to 50% of these women will show clinical signs of hyperandrogenism and 60% to 70% of female infants born to masculinized mothers will themselves exhibit some degree of virilization. [4]


  Case report Top


The present case report is about a 28 year old full-term pregnant women without any significant medical or surgical history was admitted in obstetrics ward. The abdominal ultrasonogram revealed an enlarged left ovarian mass measuring 12 cm × 7 cm × 5 cm and showed heterogeneous echogenicity. The fetus had a normal sonographic appearance. The patient underwent caesarean section. Intra-operatively surgeons found left sided ovarian mass, suspecting to be an ovarian neoplasm salpingo-oophorectomy was performed. The specimen was submitted for histopathological examination in 10% formalin.

Pathological findings

Macroscopic examination showed an enlarged ovary measuring 11 cm × 6 cm × 5 cm. Cut surface of the ovary was circumscribed with multiple nodules which was soft, fleshy and grey yellow to grey brown [Figure 1]. Microscopic sections studied showed round to polygonal cells arranged in monolayer sheets with round to oval vesicular nucleus and abundant eosinophilic granular cytoplasm [Figure 2] and [Figure 3]. There were no areas of necrosis and mitosis. Reinke crystals were not found in the sections studied; therefore Leydig cell tumor and steroid cell tumor were ruled out and final diagnosis of luteoma of pregnancy was offered.
Figure 1: Cut surface of the ovary showing multiple nodules

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Figure 2: Monolayer sheet of cells arranged in islands (H and
E, ×100)


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Figure 3: Round to polygonal cells with abundant eosinophilic
granular cytoplasm (H and E, ×400)


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  Discussion Top


Luteoma of pregnancy is a rare condition, which can mimic a solid ovarian neoplasm. [5] It most often occurs in the third and fourth decades and is associated with increased prevalence in the African American population and in the multiparous state. It is multinodular in half the cases and bilateral in a third of the cases. [6] In 1975 Garcia-Bunuel et al. in their study have reported the largest published a series of luteoma of pregnancy (20 patients). [7]

Pregnancy luteomas are variable in size, ranging from microscopic to over 20 cm in diameter. Large luteomas rarely can cause torsion, resulting in acute abdominal pain. [8] Clinically, luteomas are often silent and only discovered incidentally during peripartum surgery. [1] On gross examination, cut surfaces of luteomas are solid, soft, tan or flesh colored, with hemorrhagic foci. [7] Microscopically luteomas are sharply circumscribed nodules composed of polygonal cells arranged in sheets, cords or small clusters or they surround follicle like spaces containing colloid like material. The cytoplasm is abundant eosinophilic and finely granular. The nuclei may be slightly pleomorphic and hyperchromatic. [9] The present case was found incidentally during cesarean section and similar pathological findings were observed as discussed above. Hence, it was diagnosis as pregnancy luteoma.

The differential diagnosis for pregnancy luteomas are extensive and includes granulosa cell tumors, thecomas, Sertoli-Leydig cell tumors, pure Leydig (hilar) cell tumors, unclassified sex cord-stromal tumors, stromal hyperthecosis, stromal luteomas and hyperreactio luteinalis. However, hyperreactio luteinalis has never been reported in association with fetal virilization. [10] In our case, Reinke crystals were not found in the sections studied; therefore Leydig cell tumor and steroid cell tumor were ruled out.

During a normal pregnancy, the maternal circulating testosterone level can increase and especially in the third trimester. The serum levels of total testosterone may rise up to 7 times the non-pregnant levels and this physiological condition does not cause virilization. Virilization during pregnancy is a rare clinical event and it is most commonly caused by pregnancy luteoma or hyperreactio luteinalis. [11] since virilization was not seen in our case, hormonal studies were not done. Pregnancy luteomas typically undergo spontaneous postpartum regression, [12] usually within 3 months of delivery. Serum testosterone levels usually return to normal by 2 weeks postpartum.


  Conclusion Top


The luteoma of pregnancy is a rare condition which probably represents an unusual response to the altered hormonal environment in pregnancy and mimics either a solid or complex cystic ovarian neoplasm. It regresses in the postpartum period. A good appreciation of the clinical and imaging features of pregnancy luteoma can obviate the requirement of an unnecessary surgery or interruption of pregnancy. In difficult clinical cases with atypical presentation biopsy of this lesion with intra-operative frozen section may allow preservation of the ovary.



 
  References Top

1.
Sternberg WH, Barclay DL. Luteoma of pregnancy. Am J Obstet Gynecol 1966;95:165-84.  Back to cited text no. 1
    
2.
Wang YC, Su HY, Liu JY, Chang FW, Chen CH. Maternal and female fetal virilization caused by pregnancy luteomas. Fertil Steril 2005;84:509.  Back to cited text no. 2
    
3.
Janovski NA, Paramanandhan TL. Ovarian tumors. Tumors and tumor-like conditions of the ovaries, fallopian tubes and ligaments of the uterus. Major Probl Obstet Gynecol 1973;4:1-245.  Back to cited text no. 3
    
4.
Verhoeven AT, Mastboom JL, van Leusden HA, van der Velden WH. Virilization in pregnancy coexisting with an (ovarian) mucinous cystadenoma: A case report and review of virilizing ovarian tumors in pregnancy. Obstet Gynecol Surv 1973;28:597-622.  Back to cited text no. 4
    
5.
Sternberg WH. Nonfunctioning ovarian neoplasms. In: Grady HG, editor. The Ovary. Baltimore: William & Wilkins; 1963. p. 209.  Back to cited text no. 5
    
6.
Clement PB. Tumor-like lesions of the ovary associated with pregnancy. Int J Gynecol Pathol 1993;12:108-15.  Back to cited text no. 6
    
7.
Garcia-Bunuel R, Berek JS, Woodruff JD. Luteomas of pregnancy. Obstet Gynecol 1975;45:407-14.  Back to cited text no. 7
    
8.
Joshi R, Dunaif A. Ovarian disorders of pregnancy. Endocrinol Metab Clin North Am 1995;24:153-69.  Back to cited text no. 8
    
9.
Robert HY, Philip BC. Miscellaneous primary tumors, secondary tumors, and nonneoplastic lesions of ovary. Diagnostic Surgical Pathology, Sternberg's. 5 th ed. Lippincott Williams and Wilkins, Philadelphia; 2010. p. 2365-6.  Back to cited text no. 9
    
10.
Tinkanen H, Kuoppala T. Virilization during pregnancy caused by ovarian mucinous cystadenocarcinoma. Acta Obstet Gynecol Scand 2001;80:476-7.  Back to cited text no. 10
    
11.
Young RH, Dudley AG, Scully RE. Granulosa cell, Sertoli-Leydig cell, and unclassified sex cord-stromal tumors associated with pregnancy: A clinicopathological analysis of thirty-six cases. Gynecol Oncol 1984;18:181-205.  Back to cited text no. 11
    
12.
Wang HK, Sheu MH, Guo WY, Hong CH, Chang CY. Magnetic resonance imaging of pregnancy luteoma. J Comput Assist Tomogr 2003;27:155-7.  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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