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.
Gynaecology Case Reports 429
Figure 1. (A) Remnant left fallopian tube. Absent left ovary. Prevesical peritoneum after adhesiolysis. (B) Remnant left fallopian tube. Absent left ovary. (C) Right adnexa adherent to parietal peritoneum. Hydrosalpinx. Twisted fallopian tube. (D) Twisted tube around the ovary. Fimbria adherent to anterior abdominal wall peritoneum (prevesical peritoneum). (E) Prevesical peritoneum after lysis of the right fallopian tube adhesions.
The absence of the ipsilateral fallopian tube or a part of it was noticed in the majority of reported cases. Only one case of unilateral ovarian agenesis with normal ipsilateral tube was mentioned (Eustace 1992). When the contralateral adnexa is functional, fertility is not impaired. Gambadauro and Campo (2005) reported one spontaneous pregnancy after myomectomy in an infertile patient with unilateral ovarian agenesis. Demir and Guven (2007) reported the incidental finding of unilateral ovarian agenesis during caesarean section. Two possible explanations for unilateral ovarian agenesis are described in literature: the mechanical theory and the embryological theory. The embryological theory suggests a defect in the development of the entire Müllerian and mesonephric system on one side, or a defect localised in the region of the genital ridge and the caudal part of the Müllerian duct (Mylonas et al. 2003). In such cases, malformations of the uterus and of the urinary tract should also be present. This association of anomalies was described by Demir and Guven (2007) as a finding during caesarean delivery, and by Mulayim et al. (2003) during exploratory laparoscopy. Eustace (1992) suggested a compromised blood supply to the caudal part of the Müllerian duct, to the cells of the genital ridge or to the primitive ovary. The mechanical theory claims that a complete torsion of the ovarian pedicle is followed by ovarian and tubal necrosis (Sirisena 1978). But such an incident in adult life would be accompanied by severe pain, urging the patient to seek medical attention. Sivanesaratnam (1986) suggested that this episode could occur in utero. In support of this theory is a report by Dueck et al. (2001), who diagnosed a
cystic mass on a 37-week fetus; 8 months after delivery, the infant had surgery to remove the cyst: the left ovary could not be found (Dueck et al. 2001). A review of the literature also revealed two cases of absence of one adnexa in conjunction with a free-floating mass that could be identified as an ovary (Mylonas et al. 2003). However, a clear developmental reason for this malformation has not yet been found. This paper is particularly interesting, as it is the report of a very rare case, 1/11,240 women. Just a few other cases have been reported in literature so far. In our particular case, since no genito-urinary tract anomalies were associated, the embryological hypothesis is less probable. The chronic pelvic inflammatory disease, discovered during laparoscopy and confirmed by the result of the peritoneal washing culture, explains the abdominal pain described by the patient. The patient’s fertility was not affected by this condition, since she already had two pregnancies. The aspect of the right tube twisted around the right ovary and adherent to the anterior peritoneum suggests that such torsion might have occurred on the left side as well. However, the patient could not describe an episode of acute abdominal pain in the past. In our opinion, unilateral ovarian agenesis will be encountered more frequently in the future, because of the wider use of exploratory laparoscopy in the evaluation of patients with infertility or pelvic inflammatory disease. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
430
Gynaecology Case Reports
J Obstet Gynaecol Downloaded from informahealthcare.com by Nanyang Technological University on 08/21/15 For personal use only.
References Demir B, Guven S. 2007. An incidental finding of unicornuate uterus with unilateral ovarian agenesis during cesarean delivery. Archives of Gynecology and Obstetrics 276:91–93. Dueck A, Poenaru D, Jamieson MA et al. 2001. Unilateral ovarian agenesis and fallopian tube maldescent. Pediatric Surgery International 17:228–229. Eustace DL. 1992. Congenital absence of fallopian tube and ovary. European Journal of Obstetrics and Gynecology, and Reproductive Biology 46:157–159. Gambadauro P, Campo V. 2005. Spontaneous pregnancy following multiple laparoscopic myomectomy in a sterile patient with unilateral ovarian agenesis. European Journal of Obstetrics and Gynecology and Reproductive Biology 118:270–271. Georgy FM, Viechnicki MB. 1974. Absence of an ovary and uterine tube. Obstetrics and Gynecology 44:441–442. Mulayim B, Demirbasoglu S, Oral S. 2003. Unicornuate uterus and unilateral ovarian agenesis associated with pelvic kidney. Surgical Endoscopy 17:161. Mylonas I, Hansch S, Markmann S et al. 2003. Unilateral ovarian agenesis: report of three cases and review of the literature. Archives of Gynecology and Obstetrics 268:57–60. Rapisarda G, Pappalardo E. 2009. Unilateral ovarian and fallopian tube agenesis. Archives of Gynecology and Obstetrics 280:849–850. Sirisena LA. 1978. Unexplained absence of an ovary and uterine tube. Postgraduate Medical Journal 54:423–424. Sivanesaratnam V. 1986. Unexplained unilateral absence of ovary and fallopian tube. European Journal of Obstetrics, Gynecology, and Reproductive Biology 22:103–105.
an ectopic gestation (Mynbaev et al. 2013). The incidence is approximately 1 per 30,000 pregnancies in spontaneous conceptions, with increased risks estimated as 1:100 to 1:500 in assisted conceptions (Hassani et al. 2010).
Case report A 29-year-old multiparous woman of Caucasian origin presented at 5 weeks’ gestation with a 4-day history of severe right iliac fossa pain, nausea and vomiting. She had no vaginal bleeding. Her current pregnancy was conceived spontaneously. Clinical examination revealed a conscious woman with normal observations. Abdominal examination showed a soft abdomen and increased right iliac fossa tenderness. The blood results on admission were normal and the hCG levels were 21,838 mIU/ml. Abdominal ultrasonography (Figure 1) revealed a viable intrauterine gestation of 5 weeks, a solid mass in the right adnexa and free fluid in the pelvis. The patient underwent diagnostic laparoscopy, which confirmed right tubal ectopic pregnancy, therefore a right salpingectomy was performed. The postoperative period was uneventful. Histology of the salpingectomy specimen confirmed an ectopic gestation. An abdominal ultrasound scan was performed 2 weeks postoperatively, showing a viable intrauterine pregnancy, which proceeded without complications to a full-term normal delivery.
Discussion
A case of heterotopic pregnancy following spontaneous conception E. Tingi, J. Rowland & L. Hanna Department of Obstetrics and Gynaecology, Tameside General Hospital, Ashton-under-Lyne, UK DOI: 10.3109/01443615.2014.958445 Correspondence: E. Tingi, Department of Obstetrics and Gynaecology, Tameside General Hospital, Fountain Street, Ashton-under-Lyne OL6 9RW, UK. E-mail: [email protected]
Introduction Heterotopic pregnancy is defined as the presence of multiple pregnancies with one or more intrauterine pregnancies co-existing with
The commonest risk factors for ectopic pregnancy include the use of intrauterine devices, pelvic adhesions, previous tubal surgery, pelvic inflammatory disease and assisted conception (Borna et al. 2007). In our case, the patient did not have any risk factors for ectopic pregnancy. Heterotopic pregnancies are diagnosed at 5–34 weeks’ gestation (Hassani et al. 2010) and they can be missed very easily in the presence of intrauterine pregnancy. It has been reported that 70% of the heterotopic pregnancies are diagnosed between 5 and 8 weeks’ gestation; 20% between 9 and 10 weeks; and 10% after the 11th week (Tal et al. 1996). The diagnostic role of βhCG (human chorionic gonadotropin) levels in heterotopic pregnancy is debatable (Umranikar et al. 2009). The detection rate of heterotopic pregnancy with transvaginal ultrasound scans can vary from 41% to 84% (Umranikar et al. 2009). The easy access for early ultrasound scan for patients with previous ectopic pregnancy, as well as for patients who underwent assisted conceptions, may have important impact on the early diagnosis of heterotopic pregnancy. However, women with spontaneous conception do not necessarily have early ultrasound scans (Umranikar et al. 2009).
Figure 1. Viable intrauterine gestation and a solid mass in the right adnexa; suspected ectopic pregnancy.
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.
Gynaecology Case Reports 429
Figure 1. (A) Remnant left fallopian tube. Absent left ovary. Prevesical peritoneum after adhesiolysis. (B) Remnant left fallopian tube. Absent left ovary. (C) Right adnexa adherent to parietal peritoneum. Hydrosalpinx. Twisted fallopian tube. (D) Twisted tube around the ovary. Fimbria adherent to anterior abdominal wall peritoneum (prevesical peritoneum). (E) Prevesical peritoneum after lysis of the right fallopian tube adhesions.
The absence of the ipsilateral fallopian tube or a part of it was noticed in the majority of reported cases. Only one case of unilateral ovarian agenesis with normal ipsilateral tube was mentioned (Eustace 1992). When the contralateral adnexa is functional, fertility is not impaired. Gambadauro and Campo (2005) reported one spontaneous pregnancy after myomectomy in an infertile patient with unilateral ovarian agenesis. Demir and Guven (2007) reported the incidental finding of unilateral ovarian agenesis during caesarean section. Two possible explanations for unilateral ovarian agenesis are described in literature: the mechanical theory and the embryological theory. The embryological theory suggests a defect in the development of the entire Müllerian and mesonephric system on one side, or a defect localised in the region of the genital ridge and the caudal part of the Müllerian duct (Mylonas et al. 2003). In such cases, malformations of the uterus and of the urinary tract should also be present. This association of anomalies was described by Demir and Guven (2007) as a finding during caesarean delivery, and by Mulayim et al. (2003) during exploratory laparoscopy. Eustace (1992) suggested a compromised blood supply to the caudal part of the Müllerian duct, to the cells of the genital ridge or to the primitive ovary. The mechanical theory claims that a complete torsion of the ovarian pedicle is followed by ovarian and tubal necrosis (Sirisena 1978). But such an incident in adult life would be accompanied by severe pain, urging the patient to seek medical attention. Sivanesaratnam (1986) suggested that this episode could occur in utero. In support of this theory is a report by Dueck et al. (2001), who diagnosed a
cystic mass on a 37-week fetus; 8 months after delivery, the infant had surgery to remove the cyst: the left ovary could not be found (Dueck et al. 2001). A review of the literature also revealed two cases of absence of one adnexa in conjunction with a free-floating mass that could be identified as an ovary (Mylonas et al. 2003). However, a clear developmental reason for this malformation has not yet been found. This paper is particularly interesting, as it is the report of a very rare case, 1/11,240 women. Just a few other cases have been reported in literature so far. In our particular case, since no genito-urinary tract anomalies were associated, the embryological hypothesis is less probable. The chronic pelvic inflammatory disease, discovered during laparoscopy and confirmed by the result of the peritoneal washing culture, explains the abdominal pain described by the patient. The patient’s fertility was not affected by this condition, since she already had two pregnancies. The aspect of the right tube twisted around the right ovary and adherent to the anterior peritoneum suggests that such torsion might have occurred on the left side as well. However, the patient could not describe an episode of acute abdominal pain in the past. In our opinion, unilateral ovarian agenesis will be encountered more frequently in the future, because of the wider use of exploratory laparoscopy in the evaluation of patients with infertility or pelvic inflammatory disease. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
430
Gynaecology Case Reports
J Obstet Gynaecol Downloaded from informahealthcare.com by Nanyang Technological University on 08/21/15 For personal use only.
References Demir B, Guven S. 2007. An incidental finding of unicornuate uterus with unilateral ovarian agenesis during cesarean delivery. Archives of Gynecology and Obstetrics 276:91–93. Dueck A, Poenaru D, Jamieson MA et al. 2001. Unilateral ovarian agenesis and fallopian tube maldescent. Pediatric Surgery International 17:228–229. Eustace DL. 1992. Congenital absence of fallopian tube and ovary. European Journal of Obstetrics and Gynecology, and Reproductive Biology 46:157–159. Gambadauro P, Campo V. 2005. Spontaneous pregnancy following multiple laparoscopic myomectomy in a sterile patient with unilateral ovarian agenesis. European Journal of Obstetrics and Gynecology and Reproductive Biology 118:270–271. Georgy FM, Viechnicki MB. 1974. Absence of an ovary and uterine tube. Obstetrics and Gynecology 44:441–442. Mulayim B, Demirbasoglu S, Oral S. 2003. Unicornuate uterus and unilateral ovarian agenesis associated with pelvic kidney. Surgical Endoscopy 17:161. Mylonas I, Hansch S, Markmann S et al. 2003. Unilateral ovarian agenesis: report of three cases and review of the literature. Archives of Gynecology and Obstetrics 268:57–60. Rapisarda G, Pappalardo E. 2009. Unilateral ovarian and fallopian tube agenesis. Archives of Gynecology and Obstetrics 280:849–850. Sirisena LA. 1978. Unexplained absence of an ovary and uterine tube. Postgraduate Medical Journal 54:423–424. Sivanesaratnam V. 1986. Unexplained unilateral absence of ovary and fallopian tube. European Journal of Obstetrics, Gynecology, and Reproductive Biology 22:103–105.
an ectopic gestation (Mynbaev et al. 2013). The incidence is approximately 1 per 30,000 pregnancies in spontaneous conceptions, with increased risks estimated as 1:100 to 1:500 in assisted conceptions (Hassani et al. 2010).
Case report A 29-year-old multiparous woman of Caucasian origin presented at 5 weeks’ gestation with a 4-day history of severe right iliac fossa pain, nausea and vomiting. She had no vaginal bleeding. Her current pregnancy was conceived spontaneously. Clinical examination revealed a conscious woman with normal observations. Abdominal examination showed a soft abdomen and increased right iliac fossa tenderness. The blood results on admission were normal and the hCG levels were 21,838 mIU/ml. Abdominal ultrasonography (Figure 1) revealed a viable intrauterine gestation of 5 weeks, a solid mass in the right adnexa and free fluid in the pelvis. The patient underwent diagnostic laparoscopy, which confirmed right tubal ectopic pregnancy, therefore a right salpingectomy was performed. The postoperative period was uneventful. Histology of the salpingectomy specimen confirmed an ectopic gestation. An abdominal ultrasound scan was performed 2 weeks postoperatively, showing a viable intrauterine pregnancy, which proceeded without complications to a full-term normal delivery.
Discussion
A case of heterotopic pregnancy following spontaneous conception E. Tingi, J. Rowland & L. Hanna Department of Obstetrics and Gynaecology, Tameside General Hospital, Ashton-under-Lyne, UK DOI: 10.3109/01443615.2014.958445 Correspondence: E. Tingi, Department of Obstetrics and Gynaecology, Tameside General Hospital, Fountain Street, Ashton-under-Lyne OL6 9RW, UK. E-mail: [email protected]
Introduction Heterotopic pregnancy is defined as the presence of multiple pregnancies with one or more intrauterine pregnancies co-existing with
The commonest risk factors for ectopic pregnancy include the use of intrauterine devices, pelvic adhesions, previous tubal surgery, pelvic inflammatory disease and assisted conception (Borna et al. 2007). In our case, the patient did not have any risk factors for ectopic pregnancy. Heterotopic pregnancies are diagnosed at 5–34 weeks’ gestation (Hassani et al. 2010) and they can be missed very easily in the presence of intrauterine pregnancy. It has been reported that 70% of the heterotopic pregnancies are diagnosed between 5 and 8 weeks’ gestation; 20% between 9 and 10 weeks; and 10% after the 11th week (Tal et al. 1996). The diagnostic role of βhCG (human chorionic gonadotropin) levels in heterotopic pregnancy is debatable (Umranikar et al. 2009). The detection rate of heterotopic pregnancy with transvaginal ultrasound scans can vary from 41% to 84% (Umranikar et al. 2009). The easy access for early ultrasound scan for patients with previous ectopic pregnancy, as well as for patients who underwent assisted conceptions, may have important impact on the early diagnosis of heterotopic pregnancy. However, women with spontaneous conception do not necessarily have early ultrasound scans (Umranikar et al. 2009).
Figure 1. Viable intrauterine gestation and a solid mass in the right adnexa; suspected ectopic pregnancy.
