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Coexistence of a round ligament dermoid cyst and struma ovarii in pregnancy

2006; Elsevier BV; Volume: 126; Issue: 2 Linguagem: Inglês

10.1016/j.ejogrb.2005.11.045

1872-7654

Ihab M. Usta, Nabil G. Khoury, Ali Khalil, Anwar H. Nassar,

Testicular diseases and treatments

We report on a 33-year-old G2P1 at 385/7 weeks of gestation who was admitted for an elective cesarean section due to a previous posterior colpoperineorrhaphy. Three months prior to pregnancy she underwent bilateral laparoscopic ovarian cystectomies (3 cm × 4 cm and 3 cm × 5 cm). The cysts were found to be mature cystic teratomas. At the time of the laparoscopy, a 3 cm × 2 cm left round ligament mass was noted but was left intact because it was thought to be a fibroid (Fig. 1). Her antenatal course was remarkable for the persistence of the previously detected mass on a first trimester ultrasound. It still measured 3 cm × 2 cm and was of a heterogeneous echogenicity located on the left side of the pelvis independent of the ipsilateral ovary. After the delivery of a live healthy female newborn weighing 3530 g with Apgar scores of 9 and 10 at 1 and 5 min, respectively, the pelvic organs were inspected. The left round ligament 3 cm × 2 cm mass was noted to be firm, oval in shape and well delineated. During cystectomy, the mass ruptured and yellowish sebaceous material and hair were expelled. In addition, a right hard 2.5 cm × 2 cm ovarian cyst was detected and resected. This cyst was neither present during the laparoscopy 1 year ago, nor was it detected on the first trimester ultrasound. Histological examination of the pathology specimen revealed a mature cystic teratoma of the left round ligament, and struma ovarii in the right ovary. Our report is the first to describe the coexistence of two of the rarest presentations of dermoid cysts. Mature cystic teratomas, 8–15% of which are bilateral, are the most common germ cell tumor of the ovary, comprising approximately 20% of all ovarian neoplasms [ [1] Weldon-Linne C.M. Rushovich A.M. Benign ovarian cystic teratomas with homunculi. Obstet Gynecol. 1983; 61: S88-S94 PubMed Google Scholar ]. They are composed of well differentiated derivatives of the three germ layers, with ectodermal elements predominating. They are mainly described in the ovaries but extraovarian sites have been previously described including the fallopian tubes, uterus, omentum, vaginal wall, neck, thorax, and abdomen. Round ligament tumors are uncommon and include fibromyomas, adenomyomas, endometriomas, and serous cysts. A possible explanation for the development of these tumors in such unusual locations is that during migration of the germ cells into the genital ridge tissue, some cells do not reach their destination and become atretic while others survive to become germ cell tumors [ [2] Quigley M.M. Gwatkin R.B.L. Embryology and developmental defects of the female reproductive system. in: Scott J.R. DiSaia P.J. Hammon C.B. Spellacy W.N. Danforth's obstetrics and gynecology. JB Lippincott, Philadelphia1994: 11-27 Google Scholar ]. Because round ligament dermoids are rare, we performed a MEDLINE search of the English literature from 1966 to October 2004, using the keywords "round ligament," and "teratoma" or "dermoid", and only two cases of round ligament dermoids were identified. In the first, the cysts were bilateral and were discovered at cesarean section [ [3] Kaleli B. Aktan E. Bayramoglu H. Alatas E. Mature cystic teratoma in round ligament: case report. Eur J Obstet Gynecol Reprod Biol. 1997; 74: 195-196 Abstract Full Text PDF PubMed Scopus (12) Google Scholar ], and in the second it was detected during workup for infertility and was the first case to be managed laparoscopically [ [4] De Los Rios J.F. Ochoa J.G. Mejia J.M. Mesa A. Laparoscopic management of teratoma of the round ligament. J Am Assoc Gynecol Laparosc. 2004; 11: 265-268 Abstract Full Text Full Text PDF PubMed Scopus (5) Google Scholar ]. Similar to our case, both masses were mature cystic teratomas but none of these patients had contralateral struma ovarii. Although thyroid tissue has been demonstrated in 12–15% of mature cystic teratomas [ [5] Sifakis S. Panayiotides I.G. Angelakis E. Martavatzis N. Koumantakis E. Benign struma ovarii complicating pregnancy: a case report and review of the literature. Arch Gynecol Obstet. 2003; 269: 72-73 Crossref PubMed Scopus (8) Google Scholar ], struma ovarii is reserved to cysts where thyroid tissue constitutes more than 50% of the tumor. It comprises about 2% of ovarian dermoids [ [5] Sifakis S. Panayiotides I.G. Angelakis E. Martavatzis N. Koumantakis E. Benign struma ovarii complicating pregnancy: a case report and review of the literature. Arch Gynecol Obstet. 2003; 269: 72-73 Crossref PubMed Scopus (8) Google Scholar ], is usually unilateral and measures less than 10 cms. It presents mainly in the 5th decade of life and is rarely reported during pregnancy [ [6] Mancuso A. Triolo O. Leonardi I. De Vivo A. Struma ovarii: a rare benign pathology which may erroneously suggest malignancy. Acta Obstet Gynecol Scand. 2001; 80: 1075-1076 Crossref PubMed Scopus (18) Google Scholar ]. The most feared of complication is malignant transformation which occurs in 0.3% of cases [ [5] Sifakis S. Panayiotides I.G. Angelakis E. Martavatzis N. Koumantakis E. Benign struma ovarii complicating pregnancy: a case report and review of the literature. Arch Gynecol Obstet. 2003; 269: 72-73 Crossref PubMed Scopus (8) Google Scholar ]. This might warrant excision of these tumors even in young patients like ours who is much younger than the average age of occurrence of these neoplasms.