Gynaecology Case Reports 427
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Giron AM, Passerotti CC, Nguyen H et al. 2011. Bladder exstrophy: reconstructed female patients achieving normal pregnancy and delivering normal babies. International Brazilian Journal Urology 37:605–610. Ikeme AC. Pregnancy in women after repair of bladder exstrophy. 1981. Two case reports. British Journal of Obstetrics and Gynaecology 88:327–328. Mariona FG, Evans TN. 1982. Pregnancy following repair of anal and vaginal atresia and bladder exstrophy. Obstetrics and Gynecology 59: 653–655. Mathews RI, Gan M, Gearhart JP. 2003. Urogynaecological and obstetric issues in women with the exstrophy-epispadias complex. BJU International 91:845–849. Sayeeda S, Begum F, Akhter N et al. 2012. Successful pregnancy in a woman after repair of bladder exstrophy. Mymensingh Medical Journal 21:763–766. Sharma D, Singhal SR, Singhal SK. 1998. Successful pregnancy in a patient with previous bladder exstrophy. Australia New Zealand Journal of Obstetrics and Gynaecology 38:227–228. Thomas JC, Adams MC. 2009. Female sexual function and pregnancy after genitourinary reconstruction. Journal of Urology 182:2578–2584.
Laparoscopic management of a torted dermoid cyst with prostatic tissue in pregnancy S. Das1, C. Guha1, S. Honakeri2, A. Fleming2 & D. Uchil1 Departments of 1Obstetrics and Gynaecology and 2Pathology, University Hospital Lewisham, London, UK DOI: 10.3109/01443615.2014.958443 Correspondence: S. Das, Department of Obstetrics and Gynaecology, The Royal Free Hospital, Pond Street, London, NW3 2QG, UK. E-mail: sayantana_dr@ yahoo.co.in
Introduction The incidence of adnexal masses in pregnancy is around 2–10%. They are usually detected during routine antenatal scans with an incidence of 1–4% detected by ultrasound. The majority of these resolve spontaneously. Mature cystic teratoma (dermoid cyst) is one of the most common ovarian tumours and contains tissue typically representing all three germ layers. The finding of prostatic tissue in an ovarian teratoma is very rare. We describe a unique case of a pregnant woman presenting with torsion of a dermoid cyst, who underwent laparoscopic detorsion of the ovary and ovarian cystectomy. Histologically, the dermoid cyst showed the presence of prostatic tissue.
Case report A 22-year-old G2P1 presented to the Accident and Emergency department at 14 weeks’ gestation, with a history of right iliac fossa pain associated with nausea and vomiting. An urgent abdominal and pelvic scan showed an 84 ⫻ 57 ⫻ 68 mm well-defined cystic mass in the pelvis. Her blood tests, including tumour markers, were within normal limits. She was initially managed conservatively in view of her pregnancy, however at 18 weeks’ gestation, due to her persistent symptoms and suspicion of ovarian torsion, she was treated surgically. Laparoscopy using open technique entry revealed the presence of a 10 cm right ovarian cyst, which had twisted twice around its pedicle (Figure 1). The left ovary was normal and the size of the uterus corresponded to the period of gestation. The right ovary was detorted by gentle manipulation. A laparoscopic assisted cystectomy with an aim to minimise risk of spillage and shorten duration of surgery, was performed. She made an excellent recovery and was discharged home the following day. Histology revealed a mature cystic teratoma with a rare finding of prostatic tissue. Grossly, the cut surface showed a unilocular cyst lined by a tan membranous tissue containing hair. Microscopically, the cyst was focally lined by skin with adnexal structures (hair follicles, pilosebaceous glands and sweat glands), respiratory epithelium,
Figure 1. Right torted pedicle (curved blue arrow) around the dermoid cyst (thin black arrow). Gravid uterus (thick black arrow).
transitional epithelium, salivary gland tissue and mature cartilage. In addition, well-formed benign prostatic tissue with surrounding fibromuscular stroma and a possible urethral duct remnant, was identified. There was no immature component, atypia or malignancy. Immunohistochemically, the Cytokeratin 5/6 highlighted the basal cells and glands were weakly positive for both PSA and PSAP, thus confirming the presence of benign prostatic tissue. The rest of her pregnancy was uncomplicated and she delivered a live female infant vaginally at term, following induction of labour.
Discussion Benign cystic teratomas of the ovary are common neoplasms containing tissue from all three germ layers (Vadmal and Hajdu 1996). They constitute 95% of the germ cell tumours. Mature cystic teratomas account for 27–44% of all ovarian tumours and up to 58% of benign tumours (Zanetto and Downey 2011; Koonings et al. 1989). The risk of torsion in dermoid cysts is approximately 15% and occurs more frequently than other ovarian tumours. It is generally attributed to the fat content of most dermoid cysts, allowing them to float within the abdominal or pelvic cavity (Berek 2006). Adnexal masses complicate 2–10% of all pregnancies with the incidence of malignancy in the range of 0–8.5%. Histologically, 37–50% of the adnexal masses in pregnancy are dermoid cysts, making them the commonest in the group. In view of the majority of non-dermoid adnexal tumours resolving spontaneously, conservative management with serial ultrasound performed in each trimester is a reasonable option. In women delivering by caesarean section, adnexal masses should be evaluated and removed (if appropriate) during surgery. In women who deliver vaginally, a repeat scan should be performed at 6–8 weeks’ postpartum. The incidence of ovarian torsion in pregnancy varies according to the size of the adnexal mass and its relationship to the gravid uterus. In tumours ⬎ 4 cm, the incidence is almost 22%. Around 51% of torsion occur in tumours ⬎ 6 cm and 60% occur between 10 and 17 weeks’ gestation (Nick and Schmeler 2010). Ovarian torsion is a diagnostic dilemma, often confused with other acute abdominal conditions, such as acute appendicitis, cholecystitis and renal colic, especially in pregnancy, in view of its nonspecific presentation. Colour Doppler sonography showing the absence of intraparenchymal ovarian blood flow is a very useful diagnostic tool, however it has low sensitivity, missing 60% of the cases, but is highly specific. MRI is another preferred imaging modality in pregnant women, as there is reduced risk of ionising radiation (Hasiakos et al. 2008). In patients presenting with torsion (as in our case), a surgical approach is recommended. In a comparative study (Soriano et al. 1999), there were good outcomes in pregnant women who underwent either laparoscopy or laparotomy, suggesting that both procedures are safe. Patients undergoing laparoscopic surgery benefit from reduced delay in diagnosis. In addition, a panoramic view of the other
J Obstet Gynaecol Downloaded from informahealthcare.com by Nanyang Technological University on 08/21/15 For personal use only.
428
Gynaecology Case Reports
pelvic organs is obtained, thereby reducing the need for intraoperative uterine manipulation, as well as decreasing uterine irritability, which may cause miscarriage and preterm labour. Other benefits of laparoscopy are reduced perioperative pain and rapid recovery reducing analgesia requirements, paralytic ileus, thromboembolic events and infection. However, laparoscopic surgery comes with a risk of damaging the enlarged gravid uterus at the time of Veress needle or trocar insertion. This can increase risk of infection, bleeding, rupture of uterus and amniotic fluid leakage. Additionally, there is also the risk of CO2 insufflation into the uterine cavity, resulting in gas embolism and fetal hypercapnia. The above risks can be minimised by using the open technique, Palmer’s point entry or entering 6 cm above the umbilicus to allow insertion of the 10 mm infraumbilical trocar under direct vision. The ideal time for non-urgent but clinically required surgery for adnexal masses is 14–22 weeks’ gestation. As discussed earlier, the majority of these tumours are benign and hence the decision to proceed with surgery should be weighed against the risk of adverse perinatal outcomes. Miscarriage rates range from 0–4.7% and almost 12% of women experience preterm delivery (Usui et al. 2000). In this case, the histological specimen, in addition to showing the usual features of dermoid cyst showed the presence of prostatic tissue, which is a rare finding. The presence of prostatic tissue is difficult to explain, as ovarian teratomas generally have an XX chromosomal pattern, as they are derived from the parthenogenesis of germ cell, in which the second meiotic division has been suppressed by fusion of the polar bodies. The development of prostatic tissue in the absence of the Y chromosome is intriguing. Additionally, prostate is an endodermally derived organ induced by a urethral bud, which arises from the urogenital sinus, whereas the ovary is a mesodermal structure purely coelomic in nature, which lacks any developmental relationship with urogenital sinus (Lopez et al. 2008). There are three possible explanations for the presence of prostatic tissue in dermoid cysts, either from a mesodermal teratoma, a developmental anomaly or metaplasia (Halabi et al. 2002). In conclusion, finding of prostatic tissue in a mature cystic teratoma is a rare condition. This case report documents the successful laparoscopic management of a torted ovarian cyst in mid-pregnancy containing prostatic tissue with good maternal and fetal outcomes. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
References Berek JS. 2006. Benign diseases of the female reproductive tract. Berek and Novak’s gynecology. 14th ed. Philadelphia: Lippincott Williams and Wilkins. p. 473–474. Halabi M, Oliva E, Mazal PR et al. 2002. Prostatic tissue in mature cystic teratomas of the ovary: A report of four cases, including one with features of Prostatic Adenocarcinoma, and cytogenetic studies. International Journal of Gynecological Pathology 21:261–267. Hasiakos D, Papakonstantinou K, Kontoravdis A et al. 2008. Adnexal torsion during pregnancy: Report of four cases and review of the literature. Journal of Obstetrics and Gynaecology Research 34:683–687. Koonings PP, Campbell K, Mishell DR Jr et al. 1989. Relative frequency of primary ovarian neoplasms: a 10 year review. Obstetrics and Gynecology 74:921–926. Lopez JI, Santamaria IS, Claros IJ et al. 2008. Prostatic remnants in mature cystic teratoma of the ovary. Annals of Diagnostic Pathology 12:378–380. Nick AM, Schmeler K. 2010. Adnexal masses in pregnancy. Perinatology 2:13–21. Soriano D, Yefet Y, Seidman DS et al. 1999. Laparoscopy versus Laparotomy in the management of adnexal masses during pregnancy. Fertility and Sterility 71:955–960. Usui R, Minakami H, Kosuge S et al. 2000. A retrospective survey of clinical, pathologic, and prognostic features of adnexal masses operated on during pregnancy. Journal of Obstetrics and Gynaecology Research 26:89–93. Vadmal M, Hajdu SI. 1996. Prostatic tissue in benign cystic ovarian teratomas. Human Pathology 27:428–429. Zanetto U, Downey G. 2011. Benign tumours of the ovary. In: Shaw RE, Luesley D, Monga A, editors. Gynaecology. 4th ed. Edinburgh: Churchill Livingstone. p 673–674.
Unilateral ovarian agenesis with partial ipsilateral tubal agenesis D. Grigoras1, L. Pirtea1, I. Sas1 & P. Matusz2 Departments of 1Obstetrics and Gynecology and 2Anatomy, University of Medicine and Pharmacy ‘ Victor Babes’ Timisoara, Romania DOI: 10.3109/01443615.2014.958444 Correspondence: L. Pirtea, Department of Obstetrics and Gynecology, University of Medicine and Pharmacy ‘Victor Babes’, Pta Murgu Eftimie, 2, Timis, Timișoara 300041, Romania. E-mail: [email protected]
Introduction Unilateral ovarian agenesis, the complete absence of an ovary, is a very rare condition (Eustace 1992; Mylonas et al. 2003). Only a few cases have been described in the literature (Rapisarda and Pappalardo 2009). It has been suggested by Sinevastaman et al. (1986) that the incidence of unilateral ovarian agenesis is one case in 11,240 females, but since most of the women with this condition are totally asymptomatic, the precise incidence is difficult to determine. Both left and right unilateral ovarian agenesis have been described. Two possible causes are mentioned in the literature: congenital absence of one ovary or asymptomatic adnexal torsion (Mylonas et al. 2003).
Case report A 28-year-old woman was admitted in our clinic for moderate, persistent pain in the right iliac fossa ongoing for 3 weeks, unresponsive to anti-inflammatory and antispasmodic medication. On admission, the patient was in the 7th day of her menstrual cycle and reported regular menses. The patient had two normal vaginal deliveries and no history of abdominal surgery. Full blood count and urinalysis were in the normal range and the pregnancy test was negative. Swab tests taken from the cervix and the urethra were negative. Transvaginal ultrasound examination revealed: normal aspect of the uterus, an empty uterine cavity and an endometrial thickness of 0.3 cm. Acute appendicitis was taken into consideration, but ruled out due to the absence of peritonism signs on the right side of the abdomen, normal white blood count and normal gastrointestinal transit. Exploratory laparoscopy was performed. Intraoperative findings included: the absence of the left ovary; an incomplete left uterine tube – only the isthmic part of the tube was visualised (Figure 1A,B); adhesions of the sigmoid colon to the uterine fundus and the right tube; a right adnexa with aspect of chronic inflammatory disease and the right tube twisted around the ovary (Figure 1C), with fimbriae adherent to the pre-vesical peritoneum (Figure 1D,E). Peritoneal lavage was performed and samples for bacterial culture were taken and came back positive for Chlamydia trachomatis. The uterus and right adnexa were freed from adhesions. Chromopertubation was performed: functional right tube, absent left tube. After surgery, abdominal ultrasound examination and urography were performed in order to evaluate the urinary tract: no pathological aspects were found.
Discussion Unilateral ovarian agenesis is a very rare condition. Unilateral ovarian agenesis is rarely diagnosed, not because of its rare occurrence, but because most of the time, it is a condition discovered incidentally during surgery for other pathology due to the lack of symptoms of unilateral absence of an ovary. Different authors have reported this incidental discovery while performing laparoscopy for infertility (Sivanesaratnam 1986); ovarian cyst (Sirisena 1978); abdominal pain (Dueck et al. 2001); hysterectomy for menorrhagia (Georgy and Viechnicki 1974); or caesarean section (Demir and Guven 2007). Recently, laparoscopy gained ground in many centres, and the number of exploratory laparoscopies for infertility is increasing constantly. In this situation, the discovery of unilateral ovarian agenesis in asymptomatic women could become more frequent.
J Obstet Gynaecol Downloaded from informahealthcare.com by Nanyang Technological University on 08/21/15 For personal use only.
Giron AM, Passerotti CC, Nguyen H et al. 2011. Bladder exstrophy: reconstructed female patients achieving normal pregnancy and delivering normal babies. International Brazilian Journal Urology 37:605–610. Ikeme AC. Pregnancy in women after repair of bladder exstrophy. 1981. Two case reports. British Journal of Obstetrics and Gynaecology 88:327–328. Mariona FG, Evans TN. 1982. Pregnancy following repair of anal and vaginal atresia and bladder exstrophy. Obstetrics and Gynecology 59: 653–655. Mathews RI, Gan M, Gearhart JP. 2003. Urogynaecological and obstetric issues in women with the exstrophy-epispadias complex. BJU International 91:845–849. Sayeeda S, Begum F, Akhter N et al. 2012. Successful pregnancy in a woman after repair of bladder exstrophy. Mymensingh Medical Journal 21:763–766. Sharma D, Singhal SR, Singhal SK. 1998. Successful pregnancy in a patient with previous bladder exstrophy. Australia New Zealand Journal of Obstetrics and Gynaecology 38:227–228. Thomas JC, Adams MC. 2009. Female sexual function and pregnancy after genitourinary reconstruction. Journal of Urology 182:2578–2584.
Laparoscopic management of a torted dermoid cyst with prostatic tissue in pregnancy S. Das1, C. Guha1, S. Honakeri2, A. Fleming2 & D. Uchil1 Departments of 1Obstetrics and Gynaecology and 2Pathology, University Hospital Lewisham, London, UK DOI: 10.3109/01443615.2014.958443 Correspondence: S. Das, Department of Obstetrics and Gynaecology, The Royal Free Hospital, Pond Street, London, NW3 2QG, UK. E-mail: sayantana_dr@ yahoo.co.in
Introduction The incidence of adnexal masses in pregnancy is around 2–10%. They are usually detected during routine antenatal scans with an incidence of 1–4% detected by ultrasound. The majority of these resolve spontaneously. Mature cystic teratoma (dermoid cyst) is one of the most common ovarian tumours and contains tissue typically representing all three germ layers. The finding of prostatic tissue in an ovarian teratoma is very rare. We describe a unique case of a pregnant woman presenting with torsion of a dermoid cyst, who underwent laparoscopic detorsion of the ovary and ovarian cystectomy. Histologically, the dermoid cyst showed the presence of prostatic tissue.
Case report A 22-year-old G2P1 presented to the Accident and Emergency department at 14 weeks’ gestation, with a history of right iliac fossa pain associated with nausea and vomiting. An urgent abdominal and pelvic scan showed an 84 ⫻ 57 ⫻ 68 mm well-defined cystic mass in the pelvis. Her blood tests, including tumour markers, were within normal limits. She was initially managed conservatively in view of her pregnancy, however at 18 weeks’ gestation, due to her persistent symptoms and suspicion of ovarian torsion, she was treated surgically. Laparoscopy using open technique entry revealed the presence of a 10 cm right ovarian cyst, which had twisted twice around its pedicle (Figure 1). The left ovary was normal and the size of the uterus corresponded to the period of gestation. The right ovary was detorted by gentle manipulation. A laparoscopic assisted cystectomy with an aim to minimise risk of spillage and shorten duration of surgery, was performed. She made an excellent recovery and was discharged home the following day. Histology revealed a mature cystic teratoma with a rare finding of prostatic tissue. Grossly, the cut surface showed a unilocular cyst lined by a tan membranous tissue containing hair. Microscopically, the cyst was focally lined by skin with adnexal structures (hair follicles, pilosebaceous glands and sweat glands), respiratory epithelium,
Figure 1. Right torted pedicle (curved blue arrow) around the dermoid cyst (thin black arrow). Gravid uterus (thick black arrow).
transitional epithelium, salivary gland tissue and mature cartilage. In addition, well-formed benign prostatic tissue with surrounding fibromuscular stroma and a possible urethral duct remnant, was identified. There was no immature component, atypia or malignancy. Immunohistochemically, the Cytokeratin 5/6 highlighted the basal cells and glands were weakly positive for both PSA and PSAP, thus confirming the presence of benign prostatic tissue. The rest of her pregnancy was uncomplicated and she delivered a live female infant vaginally at term, following induction of labour.
Discussion Benign cystic teratomas of the ovary are common neoplasms containing tissue from all three germ layers (Vadmal and Hajdu 1996). They constitute 95% of the germ cell tumours. Mature cystic teratomas account for 27–44% of all ovarian tumours and up to 58% of benign tumours (Zanetto and Downey 2011; Koonings et al. 1989). The risk of torsion in dermoid cysts is approximately 15% and occurs more frequently than other ovarian tumours. It is generally attributed to the fat content of most dermoid cysts, allowing them to float within the abdominal or pelvic cavity (Berek 2006). Adnexal masses complicate 2–10% of all pregnancies with the incidence of malignancy in the range of 0–8.5%. Histologically, 37–50% of the adnexal masses in pregnancy are dermoid cysts, making them the commonest in the group. In view of the majority of non-dermoid adnexal tumours resolving spontaneously, conservative management with serial ultrasound performed in each trimester is a reasonable option. In women delivering by caesarean section, adnexal masses should be evaluated and removed (if appropriate) during surgery. In women who deliver vaginally, a repeat scan should be performed at 6–8 weeks’ postpartum. The incidence of ovarian torsion in pregnancy varies according to the size of the adnexal mass and its relationship to the gravid uterus. In tumours ⬎ 4 cm, the incidence is almost 22%. Around 51% of torsion occur in tumours ⬎ 6 cm and 60% occur between 10 and 17 weeks’ gestation (Nick and Schmeler 2010). Ovarian torsion is a diagnostic dilemma, often confused with other acute abdominal conditions, such as acute appendicitis, cholecystitis and renal colic, especially in pregnancy, in view of its nonspecific presentation. Colour Doppler sonography showing the absence of intraparenchymal ovarian blood flow is a very useful diagnostic tool, however it has low sensitivity, missing 60% of the cases, but is highly specific. MRI is another preferred imaging modality in pregnant women, as there is reduced risk of ionising radiation (Hasiakos et al. 2008). In patients presenting with torsion (as in our case), a surgical approach is recommended. In a comparative study (Soriano et al. 1999), there were good outcomes in pregnant women who underwent either laparoscopy or laparotomy, suggesting that both procedures are safe. Patients undergoing laparoscopic surgery benefit from reduced delay in diagnosis. In addition, a panoramic view of the other
J Obstet Gynaecol Downloaded from informahealthcare.com by Nanyang Technological University on 08/21/15 For personal use only.
428
Gynaecology Case Reports
pelvic organs is obtained, thereby reducing the need for intraoperative uterine manipulation, as well as decreasing uterine irritability, which may cause miscarriage and preterm labour. Other benefits of laparoscopy are reduced perioperative pain and rapid recovery reducing analgesia requirements, paralytic ileus, thromboembolic events and infection. However, laparoscopic surgery comes with a risk of damaging the enlarged gravid uterus at the time of Veress needle or trocar insertion. This can increase risk of infection, bleeding, rupture of uterus and amniotic fluid leakage. Additionally, there is also the risk of CO2 insufflation into the uterine cavity, resulting in gas embolism and fetal hypercapnia. The above risks can be minimised by using the open technique, Palmer’s point entry or entering 6 cm above the umbilicus to allow insertion of the 10 mm infraumbilical trocar under direct vision. The ideal time for non-urgent but clinically required surgery for adnexal masses is 14–22 weeks’ gestation. As discussed earlier, the majority of these tumours are benign and hence the decision to proceed with surgery should be weighed against the risk of adverse perinatal outcomes. Miscarriage rates range from 0–4.7% and almost 12% of women experience preterm delivery (Usui et al. 2000). In this case, the histological specimen, in addition to showing the usual features of dermoid cyst showed the presence of prostatic tissue, which is a rare finding. The presence of prostatic tissue is difficult to explain, as ovarian teratomas generally have an XX chromosomal pattern, as they are derived from the parthenogenesis of germ cell, in which the second meiotic division has been suppressed by fusion of the polar bodies. The development of prostatic tissue in the absence of the Y chromosome is intriguing. Additionally, prostate is an endodermally derived organ induced by a urethral bud, which arises from the urogenital sinus, whereas the ovary is a mesodermal structure purely coelomic in nature, which lacks any developmental relationship with urogenital sinus (Lopez et al. 2008). There are three possible explanations for the presence of prostatic tissue in dermoid cysts, either from a mesodermal teratoma, a developmental anomaly or metaplasia (Halabi et al. 2002). In conclusion, finding of prostatic tissue in a mature cystic teratoma is a rare condition. This case report documents the successful laparoscopic management of a torted ovarian cyst in mid-pregnancy containing prostatic tissue with good maternal and fetal outcomes. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
References Berek JS. 2006. Benign diseases of the female reproductive tract. Berek and Novak’s gynecology. 14th ed. Philadelphia: Lippincott Williams and Wilkins. p. 473–474. Halabi M, Oliva E, Mazal PR et al. 2002. Prostatic tissue in mature cystic teratomas of the ovary: A report of four cases, including one with features of Prostatic Adenocarcinoma, and cytogenetic studies. International Journal of Gynecological Pathology 21:261–267. Hasiakos D, Papakonstantinou K, Kontoravdis A et al. 2008. Adnexal torsion during pregnancy: Report of four cases and review of the literature. Journal of Obstetrics and Gynaecology Research 34:683–687. Koonings PP, Campbell K, Mishell DR Jr et al. 1989. Relative frequency of primary ovarian neoplasms: a 10 year review. Obstetrics and Gynecology 74:921–926. Lopez JI, Santamaria IS, Claros IJ et al. 2008. Prostatic remnants in mature cystic teratoma of the ovary. Annals of Diagnostic Pathology 12:378–380. Nick AM, Schmeler K. 2010. Adnexal masses in pregnancy. Perinatology 2:13–21. Soriano D, Yefet Y, Seidman DS et al. 1999. Laparoscopy versus Laparotomy in the management of adnexal masses during pregnancy. Fertility and Sterility 71:955–960. Usui R, Minakami H, Kosuge S et al. 2000. A retrospective survey of clinical, pathologic, and prognostic features of adnexal masses operated on during pregnancy. Journal of Obstetrics and Gynaecology Research 26:89–93. Vadmal M, Hajdu SI. 1996. Prostatic tissue in benign cystic ovarian teratomas. Human Pathology 27:428–429. Zanetto U, Downey G. 2011. Benign tumours of the ovary. In: Shaw RE, Luesley D, Monga A, editors. Gynaecology. 4th ed. Edinburgh: Churchill Livingstone. p 673–674.
Unilateral ovarian agenesis with partial ipsilateral tubal agenesis D. Grigoras1, L. Pirtea1, I. Sas1 & P. Matusz2 Departments of 1Obstetrics and Gynecology and 2Anatomy, University of Medicine and Pharmacy ‘ Victor Babes’ Timisoara, Romania DOI: 10.3109/01443615.2014.958444 Correspondence: L. Pirtea, Department of Obstetrics and Gynecology, University of Medicine and Pharmacy ‘Victor Babes’, Pta Murgu Eftimie, 2, Timis, Timișoara 300041, Romania. E-mail: [email protected]
Introduction Unilateral ovarian agenesis, the complete absence of an ovary, is a very rare condition (Eustace 1992; Mylonas et al. 2003). Only a few cases have been described in the literature (Rapisarda and Pappalardo 2009). It has been suggested by Sinevastaman et al. (1986) that the incidence of unilateral ovarian agenesis is one case in 11,240 females, but since most of the women with this condition are totally asymptomatic, the precise incidence is difficult to determine. Both left and right unilateral ovarian agenesis have been described. Two possible causes are mentioned in the literature: congenital absence of one ovary or asymptomatic adnexal torsion (Mylonas et al. 2003).
Case report A 28-year-old woman was admitted in our clinic for moderate, persistent pain in the right iliac fossa ongoing for 3 weeks, unresponsive to anti-inflammatory and antispasmodic medication. On admission, the patient was in the 7th day of her menstrual cycle and reported regular menses. The patient had two normal vaginal deliveries and no history of abdominal surgery. Full blood count and urinalysis were in the normal range and the pregnancy test was negative. Swab tests taken from the cervix and the urethra were negative. Transvaginal ultrasound examination revealed: normal aspect of the uterus, an empty uterine cavity and an endometrial thickness of 0.3 cm. Acute appendicitis was taken into consideration, but ruled out due to the absence of peritonism signs on the right side of the abdomen, normal white blood count and normal gastrointestinal transit. Exploratory laparoscopy was performed. Intraoperative findings included: the absence of the left ovary; an incomplete left uterine tube – only the isthmic part of the tube was visualised (Figure 1A,B); adhesions of the sigmoid colon to the uterine fundus and the right tube; a right adnexa with aspect of chronic inflammatory disease and the right tube twisted around the ovary (Figure 1C), with fimbriae adherent to the pre-vesical peritoneum (Figure 1D,E). Peritoneal lavage was performed and samples for bacterial culture were taken and came back positive for Chlamydia trachomatis. The uterus and right adnexa were freed from adhesions. Chromopertubation was performed: functional right tube, absent left tube. After surgery, abdominal ultrasound examination and urography were performed in order to evaluate the urinary tract: no pathological aspects were found.
Discussion Unilateral ovarian agenesis is a very rare condition. Unilateral ovarian agenesis is rarely diagnosed, not because of its rare occurrence, but because most of the time, it is a condition discovered incidentally during surgery for other pathology due to the lack of symptoms of unilateral absence of an ovary. Different authors have reported this incidental discovery while performing laparoscopy for infertility (Sivanesaratnam 1986); ovarian cyst (Sirisena 1978); abdominal pain (Dueck et al. 2001); hysterectomy for menorrhagia (Georgy and Viechnicki 1974); or caesarean section (Demir and Guven 2007). Recently, laparoscopy gained ground in many centres, and the number of exploratory laparoscopies for infertility is increasing constantly. In this situation, the discovery of unilateral ovarian agenesis in asymptomatic women could become more frequent.
