Obstetric Case Reports 91
Case report A 34-year-old woman presented to our unit with a 24 h history of right-sided abdominal pain and vaginal bleeding. She had an uneventful total abdominal hysterectomy 2 years previously, for menorrhagia; histology confirmed complete removal of the cervix with no underlying pathology. She had a postoperative infection shortly after surgery, which resolved with antibiotics, but since then had no pain or bleeding, until this presentation. Prior to the hysterectomy, a transcervical resection of endometrium had been attempted, but was complicated by a uterine perforation. She was multiparous, having had two vaginal deliveries and two caesarean sections and had a laparoscopic sterilisation 4 years previously. On examination, all of her observations were within normal limits. On abdominal examination, she was tender on the right side, with no rebound or guarding. Fresh blood was seen on speculum and a mass palpated at the top of the vault on vaginal examination. A routine pregnancy test was performed by the gynaecology nursing staff, which was positive. A quantitative plasma β-HCG was then sent, which was 634 mIU/ml. Her pain settled with simple analgesia and she was keen to go home for a family celebration. It was therefore arranged for her to return 48 h later for a pelvic ultrasound. The transvaginal ultrasound revealed a 65 mm non-vascular homogenous mass at the top of the vaginal vault, likely representing a haematoma. Both ovaries appeared normal. Repeat β-HCG at this point was 542 mIU/ml. She was treated for a presumed ectopic pregnancy, with single dose methotrexate, and 2 weeks later, her β-HCG had fallen to 7 mIU/ml. However, she presented 4 weeks later with ongoing abdominal pain. Her β-HCG was re-checked and was 2 mIU/ml. Repeat transvaginal ultrasound demonstrated the vault mass was unchanged in size or appearance, but there was also a new finding of a 39 ⫻ 62 ⫻ 44 mm cystic structure with multiple septae, within the right adnexa. She was subsequently taken to theatre for laparotomy, removal of haematoma and right salpingo-oophorectomy. Histology findings were of a cystic ovary measuring 35 ⫻ 25 ⫻ 10 mm and fallopian tube measuring 20 ⫻ 8 mm. There was fibrous tissue around the ovary with focal haemorrhage in-keeping with a haemorrhagic corpus luteum. There was no evidence of malignancy and no trophoblastic material identified. The lack of trophoblastic material was likely due to resolution of the ectopic following methotrexate therapy and any potential subtle remaining tissue may have been discarded at the time of operation as it would have looked like a clot.
fistula formation. There is also an increased risk with subtotal hysterectomy due to the remaining cervical stump, particularly if it is not adequately closed with peritonisation (Brown et al. 2002). The case presented here was of a late ectopic pregnancy, as it was over 2 years since her total abdominal hysterectomy. It is interesting that she developed postoperative sepsis following her hysterectomy (with no definite source of infection identified) and so there is a possibility there was a vaginal cuff infection, increasing the risk of fistula formation. Other cases of ectopics post-hysterectomy were generally managed surgically, however one case report described successfully using single-dose methotrexate to manage a presumed ectopic pregnancy post-hysterectomy in a haemodynamically stable woman with a β-HCG of 473 mIU/ml (Randy and McCool 2012).
Conclusion Ectopic pregnancy after hysterectomy is a very rare but potentially lifethreatening event. Prompt diagnosis can reduce morbidity and mortality. Therefore, a pregnancy test should always be performed when assessing a woman of childbearing age with abdominal pain, including women who have had a hysterectomy without oophorectomy. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
References Brown WD, Burrows L, Todd CS. 2002. Ectopic pregnancy after cesarean hysterectomy. Obstetrics and Gynecology 99:933–934. Centre for Maternal and Child Enquiries (CMACE). 2011. Saving mothers’ lives: reviewing maternal deaths to make motherhood safer: 2006–2008. The Eighth Report on Confidential Enquiries into Maternal Deaths in the United Kingdom. British Journal of Obstetrics and Gynaecology 118(Suppl 1):1–203. Friedman AM, Martin B, Matteson KA. 2013. Ectopic pregnancy after hysterectomy: a case report. Journal of Reproductive Medicine 58:75–76. Fylstra DL. 2010. Ectopic pregnancy after hysterectomy: a review and insight into etiology and prevention. Fertility and Sterility 94:431–435. Isaacs JD, Cesare CD, Cowan BD. 1996. Ectopic pregnancy following hysterectomy: an update for the 1990s. Obstetrics and Gynecology 88:732. Randy A, McCool MD. 2012. Successful medical management of ectopic pregnancy after prior hysterectomy: a case report. Journal of Reproductive Medicine 57:510. Saad Aldin E, Saadeh J, Ghulmiyyah L et al. 2012. Review article: late posthysterectomy ectopic pregnancy. Emergency Medicine Australasia 24:239–243.
Discussion Ectopic pregnancy following hysterectomy is a rare, but potentially life-threatening event. It was first described in 1895 and since then, 59 cases have been reported in the literature (Friedman et al. 2013; Fylstra 2010; Saad Alin et al. 2012). There is a much higher mortality associated with ectopic pregnancies post-hysterectomy than in women with intact uteri. Less than 0.02% of all ectopic pregnancies result in maternal death in the UK (CMACE 2011), compared with a maternal mortality rate of 7.4% for ectopics post-hysterectomy. A likely reason for this increased mortality is that the diagnosis of ectopic pregnancy is often made late, at which point, the majority have already ruptured (Saad Alin et al. 2012). Ectopic pregnancy following hysterectomy can be described as ‘early’, i.e. in the immediate postoperative period, when fertilisation likely happened prior to hysterectomy; or ‘late’, i.e. after the immediate postoperative period, when fertilisation likely occurred following the development of a vaginal-peritoneal fistula or prolapsed fallopian tube. A total of 28 of the 59 cases identified in the literature were late ectopics (Friedman et al. 2013; Saad Alin et al. 2012). Late ectopic pregnancy appears to be more common after vaginal hysterectomy or laparoscopically assisted hysterectomy, which may be related to a surgical technique of incorporating the pedicle containing the fallopian tube into the peritoneal closure, which lies next to the vagina, making fistula development or fallopian tube prolapse more likely (Isaacs et al. 1996). Infection or haematoma at the vaginal cuff are also thought to be associated with an increased risk of subsequent ectopic by increasing the chance of
Conservative surgical treatment for caesarean scar pregnancy K. Kai1, K. Shimamoto2, H. Matsumoto1 & H. Narahara1 Department of Obstetrics and Gynecology, 1Faculty of Medicine, Oita University and 2Beppu Medical Center, Oita, Japan DOI: 10.3109/01443615.2013.832179 Correspondence: K. Kai, Department of Obstetrics and Gynecology, Faculty of Medicine, Oita University, 1–1 Idaigaoka, Hasama-machi, Yufu, Oita 879–5593, Japan. E-mail: [email protected]
Introduction Caesarean scar pregnancy (CSP) is very rare, but is increasing in frequency, with an increase in caesarean section deliveries (Rotas et al. 2006). Treatment may be with combined systemic and intragestational sac methotrexate (MTX) or surgery, but there is currently no standard treatment protocol. MTX treatment has an efficacy of nearly 80% (Jurkovic et al. 2003), but requires extended surveillance of human chorionic gonadotropin (hCG) levels and is associated with adverse effects that are unacceptable to some patients (Rotas et al. 2006). Repeat CSP, uterine rupture and placenta accreta have been reported after non-surgical treatment. However, the appropriateness
92
Obstetric Case Reports scissors, followed by uterine curettage via the incision. The scar was repaired with two layers of interrupted sutures (1-0 Polysorb; Covidien, Mansfield, MA). No additional interventions were required. The clinical characteristics of the six patients are shown in Table I. The operation time was 89.5 ⫾ 48.1 min; intraoperative blood loss was 215.0 ⫾ 145.7 ml and postoperative time to normal serum hCG levels was 30.7 ⫾ 11.3 days. Histological examination of the specimens (haematoxylin and eosin staining) showed chorionic villi and discontinuity of the myometrium. Immunohistochemical examination showed myometrial invasion with intermediate trophoblast cells that were cytokeratin (CAM 5.2) positive, human placental lactogen positive and hCG negative. Curettage specimens showed no products of conception. Two patients subsequently conceived: one miscarried at 10 weeks’ gestation, and the other was delivered by elective caesarean section at 37 weeks’ gestation without complications. The other patients did not report using contraception, but it is unknown whether they tried to conceive or not.
Figure 1. Operative view of caesarean scar pregnancy (CSP) (left side, caudal; right side, cephalic). The arrow shows the protruding gestational sac.
of primary surgical treatment is still debated. We present our series of patients with CSP who underwent primary surgical resection of the gestational sac and repair of the caesarean scar, and discuss the appropriateness of this treatment.
Case series Review of the databases of two tertiary referral obstetric units from 2006 to 2012 identified eight patients who were treated for CSP. Diagnosis was by high-resolution colour Doppler vaginal ultrasonography and quantitative total serum hCG measurement, and was confirmed by magnetic resonance imaging (MRI). Ultrasonographic criteria for diagnosis were: trophoblast, mainly between the bladder and the anterior uterine wall; no fetal pole in the uterine cavity and discontinuity in the anterior uterine wall on a mid-sagittal view (Vial et al. 2000). Two patients were excluded: one initially underwent 8 days of expectant management, resulting in rupture of the ectopic lesion with massive intraperitoneal bleeding that required emergency surgery; the other had a low serum hCG level for 6 weeks’ gestation (240 mIU/ml) and no identifiable embryonic structures in the gestational sac on ultrasonography, suggesting incomplete abortion. The remaining six patients with viable CSP who did not receive any preceding treatment were included in this series. According to the policies of the institutional ethics committees, no approval was required for this retrospective review. Patients underwent laparotomy under general anaesthesia, via a vertical midline lower abdominal incision. The ectopic mass was separate from the bladder in all cases. The protruding uterine serosa was incised transversely. Scissors were inserted between the serosa and the ectopic mass and pushed laterally to each side. Extension of the incision caused the ectopic mass to bulge out (Figure 1), and the gestational sac and placental tissues were removed. The scar tissue was identified by inspection and direct palpation, and was debrided with
Discussion Primary surgical treatment options for CSP in patients who wish to preserve fertility include uterine curettage, hysteroscopic resection, laparoscopic resection and laparotomy with resection. Curettage risks severe haemorrhage, resulting from gestational sac rupture and myometrial disruption. Hysteroscopic and laparoscopic resection are suitable only in stable patients, when appropriate facilities and trained surgeons are available (Deans and Abbott 2010; Wang et al. 2006). Conversely, laparotomy is suitable for any size of ectopic mass, does not require specialised surgical skills and enables direct palpation of myometrial thickness and consistency for easy identification and debridement of the uterine scar. Primary treatment of CSP with laparotomy for resection of the gestational sac and repair of the uterine scar has only been described in six cases in the English literature (Vial et al. 2000; Coniglio and Dickinson 2004; Maymon et al. 2004; Seow et al. 2004; Shih 2004; Tagore et al. 2010). Treatment was successful in all patients. To date, there have been no reports of uterine dehiscence, recurrent CSP or placenta accreta in patients who have undergone this treatment. In conclusion, treatment of CSP with laparotomy has the following advantages: (1) suitable for any size of ectopic mass; (2) short posttreatment surveillance time; (3) adequate tissue specimens for pathological examination; (4) opportunity to repair the uterine scar; (5) preservation of fertility. We therefore consider laparotomy for resection of the gestational sac and repair of the uterine scar to be a suitable primary treatment option in patients who wish to preserve fertility. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
References Coniglio C, Dickinson JE. 2004. Pregnancy following prior Caesarean scar pregnancy rupture: Lessons for modern obstetric practice. Australian and New Zealand Journal of Obstetrics and Gynaecology 44:162–165.
Table I. Clinical characteristics of six patients with caesarean scar pregnancy. Case 1 2 3 4 5 6 Mean ⫾ SD
Age (years)
No. of previous caesarean sections
Gestational age (weeks)
Serum hCG (mIU/ml)
Operation time (min)
Blood loss (ml)
hCG clearance time (days)
Subsequent pregnancy
30 34 33 38 31 33 33.2 ⫾ 2.8
1 3 1 2 1 3 1.8 ⫾ 1.0
Unknown 10 9 8 6 7
7,240 12,320 102,700 39,140 32,500 70,000 43,983.3 ⫾ 36,421.1
96 70 77 42 181 71 89.5 ⫾ 48.1
400 320 310 50 120 90 215.0 ⫾ 145.7
17 35 18 45 39 30 30.7 ⫾ 11.3
No Yes∗ Yes† No No No
hCG, human chorionic gonadotropin; SD, standard deviation. ∗Conception at 7 months after surgery, miscarriage at 10 weeks’ gestation. †Conception at 5 months after surgery, caesarean section delivery at 37 weeks’ gestation.
Obstetric Case Reports 93 Deans R, Abbott J. 2010. Hysteroscopic management of cesarean scar ectopic pregnancy. Fertility and Sterility 93:1735–1740. Jurkovic D, Hillaby K, Woelfer B et al. 2003. First-trimester diagnosis and management of pregnancies implanted into the lower uterine segment Cesarean section scar. Ultrasound in Obstetrics and Gynecology 21:220–227. Maymon R, Halperin R, Mendlovic S et al. 2004. Ectopic pregnancies in Caesarean section scars: the 8 year experience of one medical centre. Human Reproduction 19:278–284. Rotas MA, Haberman S, Levgur M. 2006. Cesarean scar ectopic pregnancies: etiology, diagnosis, and management. Obstetrics and Gynecology 107: 1373–1381. Seow KM, Huang LW, Lin YH et al. 2004. Cesarean scar pregnancy: issues in management. Ultrasound in Obstetrics and Gynecology 23:247–253. Shih JC. 2004. Cesarean scar pregnancy: diagnosis with three-dimensional (3D) ultrasound and 3D power Doppler. Ultrasound in Obstetrics and Gynecology 23:306–307. Tagore S, Teo SH, Chua SY et al. 2010. A retrospective review of uterine scar pregnancies: single centre experience. Archives of Gynecology and Obstetrics 282:711–715. Vial Y, Petignat P, Hohlfeld P. 2000. Pregnancy in a cesarean scar. Ultrasound in Obstetrics and Gynecology 16:592–593. Wang YL, Su TH, Chen HS. 2006. Operative laparoscopy for unruptured ectopic pregnancy in a caesarean scar. British Journal of Obstetrics and Gynaecology 113:1035–1038.
Pivotal points in interstitial pregnancy: New insights in conservative medical treatment of non-ruptured interstitial pregnancy A. Pellegrino1, G. R. Damiani1, S. Landi2, M. Tartagni3, S. Tafuri4, A. Caringella3, C. Sportelli5, M. Gaetani3 & G. Loverro3 Department of Obstetrics and Gynecology, 1Malzoni Hospital, Lecco, 2Sondrio Hospital, Sondrio, 3University of Bari, Policlinico 4Department of Biomedical Sciences, Section of Hygiene, ‘Aldo Moro’ University, Bari and 5Alessandro Manzoni Hospital, Lecco, Italy DOI: 10.3109/01443615.2013.789834 Correspondence: G. R. Damiani, Department of Obstetrics and Gynecology, Malzoni Hospital, Lecco, Italy. E-mail: [email protected]
We report the efficacy of a minimally invasive approach of the multidose protocol with methotrexate (MTX) in the management of three cases of interstitial pregnancy (IP), with elevated serum b-hCG in two cases. New considerations and management strategies are discussed. Successful termination of IP and in one case, a subsequent successful pregnancy, was achieved. The process led to the development of an enhanced understanding of diagnostic modalities and their limitations, with regard to the particular entities under discussion. We also focused attention on pivotal points and anatomical features in the management of this dangerous occurrence. Long-term results with careful follow-up were analysed by instrumental procedure. This hazardous type of ectopic pregnancy can be managed with systemic administration of MTX, also in patients with elevated β-hCG values. The present report underlines that an integrated approach in early diagnosis, multidose treatment and close follow-up, are essential forms of medical management. Keywords: Cornual pregnancy, fallopian tube, interstitial pregnancy, methotrexate, multidose protocol
Introduction Interstitial pregnancy (IP) accounts for 2–4% of tubal pregnancies and is an ectopic pregnancy located outside of the uterine cavity, in the first part of the fallopian tube, surrounded by the muscular layer of the uterus, which allows a significant expansion when it hosts a pregnancy, but delays the clinical symptoms characterised
by heavy haemorrhage and high mortality rate (2.5%) (Damario and Rock 2003; Moawad et al. 2010; Tulandi and Al-Jaroudi 2004; Walsen 1957). Early diagnosis is mandatory in order to avoid massive transfusions and detrimental effects of surgery on future fertility, like cornual wedge resection or total hysterectomy (Walker 2007). The choice of treatment is still under discussion, although preliminary data revealed that medical treatment with methotrexate (MTX) is the gold standard, as in other ectopic pregnancies (Stovall and Ling 1993).
Case report 1 A 39-year-old nulliparous primigravida, presented to our institution with 7 weeks’ amenorrhoea, with recurrent vaginal bleeding lasting for 1 week. The β-hCG value was 8,166 mUI/ml, rising to 8,971 mUI/ml after 48 h. A transvaginal scanning revealed a 23 mm gestational sac at the right superolateral portion of the uterus, corresponding to 5 weeks’ gestational age, with no signs of embryonic viability, surrounded by a thin myometrium (⬍ 5 mm), outside of the empty uterine cavity and the interstitial line sign (Figure 1). These parameters have been interpreted as suggestive of an IP. Colour Doppler evidenced rich blood flow, with an image of a ring of fire, surrounding the sac and spectral tracing revealed a low resistance pattern, characteristic of trophoblastic blood flow (Figure 2). The patient was counselled about the ectopic implantation, the associated risks and all putative treatment regimens. Considering the rising levels of β-hCG and the patient’s desire to preserve fertility, systemic MTX administration was performed, at the dose of 1 mg/kg plus folinic acid 0, 1 mg/kg on days 0,2,4,6. Close follow-up was made, monitoring β-hCG and with ultrasound. The significant decline in the levels of β-hCG was observed from the 6th to 7th day onwards (Figure 3). Ultrasound monitoring revealed a gradual reduction in the size of the ectopic gestation from 24 mm to 16 mm, with a significant synchronous reduction of blood supply on the 8th day of MTX administration (Figure 4). The β-hCG fell gradually to non-pregnant levels within 36 days. After 6 months, hysteroscopy revealed a normal uterine cavity. A normal pregnancy occurred after 8 months, ending with a caesarean section for fetal distress.
Case report 2 A 37-year-old nulliparous woman, with 9 weeks’ amenorrhoea, presented to us complaining of vaginal spotting and acute abdominal pain. A transvaginal scan showed an eccentrically gestational sac of 29 mm, with embryonic viability, located at the outer right angle of fibromatous uterus. The pregnancy appeared implanted on the right extremity, where no myomas were present. The site of the gestational sac and the presence of the interstitial line sign suggested IP. The initial β-hCG value was 24, 213 mIU/l. MTX multidose protocol was administered as described above. On the 4th day, the β-hCG value was 16, 600 mIU/l, at the end of treatment on the 8th day 12, 1,312 mIU/l (Figure 5). Ultrasounds were performed on days 2, 5, 10 and then weekly until the risk of rupture, due to persistent trophoblastic proliferation, was presumed to disappear. This risk was evaluated by Doppler flow persistence in implantation site, which disappeared after 10 days from the beginning of treatment when the β-hCG level was 14.181 mIU/l. Ultrasound confirmed the resolution of the ectopic mass, 15 days after the termination of treatment.
Case report 3 A 33-year-old nulliparous woman with 7 weeks’ amenorrhoea, after a successful cycle of IVF, presented in our institution for vaginal bleeding. Ultrasonography revealed a singleton gestation, eccentrically placed in the left utero-tubal junctional area, lying 1 cm outside the most upper lateral edge of the decidualised cavity, measuring 0.4 cm. No embryo was visualised. Spectral tracing revealed a low resistant pattern deriving from trophoblastic tissue. The initial β-hCG was 1,351 mIU/l. Multidose protocol was administered. On the 8th day, β-hCG was 1,451 mIU/l, after 1 week 115 mIU/l and after another 5 days, the β-hCG level fell to 3 mIU/l.
Discussion The surrounding muscular wall of the interstitial portion confers ability to expand higher than other parts of fallopian tube.
Case report A 34-year-old woman presented to our unit with a 24 h history of right-sided abdominal pain and vaginal bleeding. She had an uneventful total abdominal hysterectomy 2 years previously, for menorrhagia; histology confirmed complete removal of the cervix with no underlying pathology. She had a postoperative infection shortly after surgery, which resolved with antibiotics, but since then had no pain or bleeding, until this presentation. Prior to the hysterectomy, a transcervical resection of endometrium had been attempted, but was complicated by a uterine perforation. She was multiparous, having had two vaginal deliveries and two caesarean sections and had a laparoscopic sterilisation 4 years previously. On examination, all of her observations were within normal limits. On abdominal examination, she was tender on the right side, with no rebound or guarding. Fresh blood was seen on speculum and a mass palpated at the top of the vault on vaginal examination. A routine pregnancy test was performed by the gynaecology nursing staff, which was positive. A quantitative plasma β-HCG was then sent, which was 634 mIU/ml. Her pain settled with simple analgesia and she was keen to go home for a family celebration. It was therefore arranged for her to return 48 h later for a pelvic ultrasound. The transvaginal ultrasound revealed a 65 mm non-vascular homogenous mass at the top of the vaginal vault, likely representing a haematoma. Both ovaries appeared normal. Repeat β-HCG at this point was 542 mIU/ml. She was treated for a presumed ectopic pregnancy, with single dose methotrexate, and 2 weeks later, her β-HCG had fallen to 7 mIU/ml. However, she presented 4 weeks later with ongoing abdominal pain. Her β-HCG was re-checked and was 2 mIU/ml. Repeat transvaginal ultrasound demonstrated the vault mass was unchanged in size or appearance, but there was also a new finding of a 39 ⫻ 62 ⫻ 44 mm cystic structure with multiple septae, within the right adnexa. She was subsequently taken to theatre for laparotomy, removal of haematoma and right salpingo-oophorectomy. Histology findings were of a cystic ovary measuring 35 ⫻ 25 ⫻ 10 mm and fallopian tube measuring 20 ⫻ 8 mm. There was fibrous tissue around the ovary with focal haemorrhage in-keeping with a haemorrhagic corpus luteum. There was no evidence of malignancy and no trophoblastic material identified. The lack of trophoblastic material was likely due to resolution of the ectopic following methotrexate therapy and any potential subtle remaining tissue may have been discarded at the time of operation as it would have looked like a clot.
fistula formation. There is also an increased risk with subtotal hysterectomy due to the remaining cervical stump, particularly if it is not adequately closed with peritonisation (Brown et al. 2002). The case presented here was of a late ectopic pregnancy, as it was over 2 years since her total abdominal hysterectomy. It is interesting that she developed postoperative sepsis following her hysterectomy (with no definite source of infection identified) and so there is a possibility there was a vaginal cuff infection, increasing the risk of fistula formation. Other cases of ectopics post-hysterectomy were generally managed surgically, however one case report described successfully using single-dose methotrexate to manage a presumed ectopic pregnancy post-hysterectomy in a haemodynamically stable woman with a β-HCG of 473 mIU/ml (Randy and McCool 2012).
Conclusion Ectopic pregnancy after hysterectomy is a very rare but potentially lifethreatening event. Prompt diagnosis can reduce morbidity and mortality. Therefore, a pregnancy test should always be performed when assessing a woman of childbearing age with abdominal pain, including women who have had a hysterectomy without oophorectomy. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
References Brown WD, Burrows L, Todd CS. 2002. Ectopic pregnancy after cesarean hysterectomy. Obstetrics and Gynecology 99:933–934. Centre for Maternal and Child Enquiries (CMACE). 2011. Saving mothers’ lives: reviewing maternal deaths to make motherhood safer: 2006–2008. The Eighth Report on Confidential Enquiries into Maternal Deaths in the United Kingdom. British Journal of Obstetrics and Gynaecology 118(Suppl 1):1–203. Friedman AM, Martin B, Matteson KA. 2013. Ectopic pregnancy after hysterectomy: a case report. Journal of Reproductive Medicine 58:75–76. Fylstra DL. 2010. Ectopic pregnancy after hysterectomy: a review and insight into etiology and prevention. Fertility and Sterility 94:431–435. Isaacs JD, Cesare CD, Cowan BD. 1996. Ectopic pregnancy following hysterectomy: an update for the 1990s. Obstetrics and Gynecology 88:732. Randy A, McCool MD. 2012. Successful medical management of ectopic pregnancy after prior hysterectomy: a case report. Journal of Reproductive Medicine 57:510. Saad Aldin E, Saadeh J, Ghulmiyyah L et al. 2012. Review article: late posthysterectomy ectopic pregnancy. Emergency Medicine Australasia 24:239–243.
Discussion Ectopic pregnancy following hysterectomy is a rare, but potentially life-threatening event. It was first described in 1895 and since then, 59 cases have been reported in the literature (Friedman et al. 2013; Fylstra 2010; Saad Alin et al. 2012). There is a much higher mortality associated with ectopic pregnancies post-hysterectomy than in women with intact uteri. Less than 0.02% of all ectopic pregnancies result in maternal death in the UK (CMACE 2011), compared with a maternal mortality rate of 7.4% for ectopics post-hysterectomy. A likely reason for this increased mortality is that the diagnosis of ectopic pregnancy is often made late, at which point, the majority have already ruptured (Saad Alin et al. 2012). Ectopic pregnancy following hysterectomy can be described as ‘early’, i.e. in the immediate postoperative period, when fertilisation likely happened prior to hysterectomy; or ‘late’, i.e. after the immediate postoperative period, when fertilisation likely occurred following the development of a vaginal-peritoneal fistula or prolapsed fallopian tube. A total of 28 of the 59 cases identified in the literature were late ectopics (Friedman et al. 2013; Saad Alin et al. 2012). Late ectopic pregnancy appears to be more common after vaginal hysterectomy or laparoscopically assisted hysterectomy, which may be related to a surgical technique of incorporating the pedicle containing the fallopian tube into the peritoneal closure, which lies next to the vagina, making fistula development or fallopian tube prolapse more likely (Isaacs et al. 1996). Infection or haematoma at the vaginal cuff are also thought to be associated with an increased risk of subsequent ectopic by increasing the chance of
Conservative surgical treatment for caesarean scar pregnancy K. Kai1, K. Shimamoto2, H. Matsumoto1 & H. Narahara1 Department of Obstetrics and Gynecology, 1Faculty of Medicine, Oita University and 2Beppu Medical Center, Oita, Japan DOI: 10.3109/01443615.2013.832179 Correspondence: K. Kai, Department of Obstetrics and Gynecology, Faculty of Medicine, Oita University, 1–1 Idaigaoka, Hasama-machi, Yufu, Oita 879–5593, Japan. E-mail: [email protected]
Introduction Caesarean scar pregnancy (CSP) is very rare, but is increasing in frequency, with an increase in caesarean section deliveries (Rotas et al. 2006). Treatment may be with combined systemic and intragestational sac methotrexate (MTX) or surgery, but there is currently no standard treatment protocol. MTX treatment has an efficacy of nearly 80% (Jurkovic et al. 2003), but requires extended surveillance of human chorionic gonadotropin (hCG) levels and is associated with adverse effects that are unacceptable to some patients (Rotas et al. 2006). Repeat CSP, uterine rupture and placenta accreta have been reported after non-surgical treatment. However, the appropriateness
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Obstetric Case Reports scissors, followed by uterine curettage via the incision. The scar was repaired with two layers of interrupted sutures (1-0 Polysorb; Covidien, Mansfield, MA). No additional interventions were required. The clinical characteristics of the six patients are shown in Table I. The operation time was 89.5 ⫾ 48.1 min; intraoperative blood loss was 215.0 ⫾ 145.7 ml and postoperative time to normal serum hCG levels was 30.7 ⫾ 11.3 days. Histological examination of the specimens (haematoxylin and eosin staining) showed chorionic villi and discontinuity of the myometrium. Immunohistochemical examination showed myometrial invasion with intermediate trophoblast cells that were cytokeratin (CAM 5.2) positive, human placental lactogen positive and hCG negative. Curettage specimens showed no products of conception. Two patients subsequently conceived: one miscarried at 10 weeks’ gestation, and the other was delivered by elective caesarean section at 37 weeks’ gestation without complications. The other patients did not report using contraception, but it is unknown whether they tried to conceive or not.
Figure 1. Operative view of caesarean scar pregnancy (CSP) (left side, caudal; right side, cephalic). The arrow shows the protruding gestational sac.
of primary surgical treatment is still debated. We present our series of patients with CSP who underwent primary surgical resection of the gestational sac and repair of the caesarean scar, and discuss the appropriateness of this treatment.
Case series Review of the databases of two tertiary referral obstetric units from 2006 to 2012 identified eight patients who were treated for CSP. Diagnosis was by high-resolution colour Doppler vaginal ultrasonography and quantitative total serum hCG measurement, and was confirmed by magnetic resonance imaging (MRI). Ultrasonographic criteria for diagnosis were: trophoblast, mainly between the bladder and the anterior uterine wall; no fetal pole in the uterine cavity and discontinuity in the anterior uterine wall on a mid-sagittal view (Vial et al. 2000). Two patients were excluded: one initially underwent 8 days of expectant management, resulting in rupture of the ectopic lesion with massive intraperitoneal bleeding that required emergency surgery; the other had a low serum hCG level for 6 weeks’ gestation (240 mIU/ml) and no identifiable embryonic structures in the gestational sac on ultrasonography, suggesting incomplete abortion. The remaining six patients with viable CSP who did not receive any preceding treatment were included in this series. According to the policies of the institutional ethics committees, no approval was required for this retrospective review. Patients underwent laparotomy under general anaesthesia, via a vertical midline lower abdominal incision. The ectopic mass was separate from the bladder in all cases. The protruding uterine serosa was incised transversely. Scissors were inserted between the serosa and the ectopic mass and pushed laterally to each side. Extension of the incision caused the ectopic mass to bulge out (Figure 1), and the gestational sac and placental tissues were removed. The scar tissue was identified by inspection and direct palpation, and was debrided with
Discussion Primary surgical treatment options for CSP in patients who wish to preserve fertility include uterine curettage, hysteroscopic resection, laparoscopic resection and laparotomy with resection. Curettage risks severe haemorrhage, resulting from gestational sac rupture and myometrial disruption. Hysteroscopic and laparoscopic resection are suitable only in stable patients, when appropriate facilities and trained surgeons are available (Deans and Abbott 2010; Wang et al. 2006). Conversely, laparotomy is suitable for any size of ectopic mass, does not require specialised surgical skills and enables direct palpation of myometrial thickness and consistency for easy identification and debridement of the uterine scar. Primary treatment of CSP with laparotomy for resection of the gestational sac and repair of the uterine scar has only been described in six cases in the English literature (Vial et al. 2000; Coniglio and Dickinson 2004; Maymon et al. 2004; Seow et al. 2004; Shih 2004; Tagore et al. 2010). Treatment was successful in all patients. To date, there have been no reports of uterine dehiscence, recurrent CSP or placenta accreta in patients who have undergone this treatment. In conclusion, treatment of CSP with laparotomy has the following advantages: (1) suitable for any size of ectopic mass; (2) short posttreatment surveillance time; (3) adequate tissue specimens for pathological examination; (4) opportunity to repair the uterine scar; (5) preservation of fertility. We therefore consider laparotomy for resection of the gestational sac and repair of the uterine scar to be a suitable primary treatment option in patients who wish to preserve fertility. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
References Coniglio C, Dickinson JE. 2004. Pregnancy following prior Caesarean scar pregnancy rupture: Lessons for modern obstetric practice. Australian and New Zealand Journal of Obstetrics and Gynaecology 44:162–165.
Table I. Clinical characteristics of six patients with caesarean scar pregnancy. Case 1 2 3 4 5 6 Mean ⫾ SD
Age (years)
No. of previous caesarean sections
Gestational age (weeks)
Serum hCG (mIU/ml)
Operation time (min)
Blood loss (ml)
hCG clearance time (days)
Subsequent pregnancy
30 34 33 38 31 33 33.2 ⫾ 2.8
1 3 1 2 1 3 1.8 ⫾ 1.0
Unknown 10 9 8 6 7
7,240 12,320 102,700 39,140 32,500 70,000 43,983.3 ⫾ 36,421.1
96 70 77 42 181 71 89.5 ⫾ 48.1
400 320 310 50 120 90 215.0 ⫾ 145.7
17 35 18 45 39 30 30.7 ⫾ 11.3
No Yes∗ Yes† No No No
hCG, human chorionic gonadotropin; SD, standard deviation. ∗Conception at 7 months after surgery, miscarriage at 10 weeks’ gestation. †Conception at 5 months after surgery, caesarean section delivery at 37 weeks’ gestation.
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Pivotal points in interstitial pregnancy: New insights in conservative medical treatment of non-ruptured interstitial pregnancy A. Pellegrino1, G. R. Damiani1, S. Landi2, M. Tartagni3, S. Tafuri4, A. Caringella3, C. Sportelli5, M. Gaetani3 & G. Loverro3 Department of Obstetrics and Gynecology, 1Malzoni Hospital, Lecco, 2Sondrio Hospital, Sondrio, 3University of Bari, Policlinico 4Department of Biomedical Sciences, Section of Hygiene, ‘Aldo Moro’ University, Bari and 5Alessandro Manzoni Hospital, Lecco, Italy DOI: 10.3109/01443615.2013.789834 Correspondence: G. R. Damiani, Department of Obstetrics and Gynecology, Malzoni Hospital, Lecco, Italy. E-mail: [email protected]
We report the efficacy of a minimally invasive approach of the multidose protocol with methotrexate (MTX) in the management of three cases of interstitial pregnancy (IP), with elevated serum b-hCG in two cases. New considerations and management strategies are discussed. Successful termination of IP and in one case, a subsequent successful pregnancy, was achieved. The process led to the development of an enhanced understanding of diagnostic modalities and their limitations, with regard to the particular entities under discussion. We also focused attention on pivotal points and anatomical features in the management of this dangerous occurrence. Long-term results with careful follow-up were analysed by instrumental procedure. This hazardous type of ectopic pregnancy can be managed with systemic administration of MTX, also in patients with elevated β-hCG values. The present report underlines that an integrated approach in early diagnosis, multidose treatment and close follow-up, are essential forms of medical management. Keywords: Cornual pregnancy, fallopian tube, interstitial pregnancy, methotrexate, multidose protocol
Introduction Interstitial pregnancy (IP) accounts for 2–4% of tubal pregnancies and is an ectopic pregnancy located outside of the uterine cavity, in the first part of the fallopian tube, surrounded by the muscular layer of the uterus, which allows a significant expansion when it hosts a pregnancy, but delays the clinical symptoms characterised
by heavy haemorrhage and high mortality rate (2.5%) (Damario and Rock 2003; Moawad et al. 2010; Tulandi and Al-Jaroudi 2004; Walsen 1957). Early diagnosis is mandatory in order to avoid massive transfusions and detrimental effects of surgery on future fertility, like cornual wedge resection or total hysterectomy (Walker 2007). The choice of treatment is still under discussion, although preliminary data revealed that medical treatment with methotrexate (MTX) is the gold standard, as in other ectopic pregnancies (Stovall and Ling 1993).
Case report 1 A 39-year-old nulliparous primigravida, presented to our institution with 7 weeks’ amenorrhoea, with recurrent vaginal bleeding lasting for 1 week. The β-hCG value was 8,166 mUI/ml, rising to 8,971 mUI/ml after 48 h. A transvaginal scanning revealed a 23 mm gestational sac at the right superolateral portion of the uterus, corresponding to 5 weeks’ gestational age, with no signs of embryonic viability, surrounded by a thin myometrium (⬍ 5 mm), outside of the empty uterine cavity and the interstitial line sign (Figure 1). These parameters have been interpreted as suggestive of an IP. Colour Doppler evidenced rich blood flow, with an image of a ring of fire, surrounding the sac and spectral tracing revealed a low resistance pattern, characteristic of trophoblastic blood flow (Figure 2). The patient was counselled about the ectopic implantation, the associated risks and all putative treatment regimens. Considering the rising levels of β-hCG and the patient’s desire to preserve fertility, systemic MTX administration was performed, at the dose of 1 mg/kg plus folinic acid 0, 1 mg/kg on days 0,2,4,6. Close follow-up was made, monitoring β-hCG and with ultrasound. The significant decline in the levels of β-hCG was observed from the 6th to 7th day onwards (Figure 3). Ultrasound monitoring revealed a gradual reduction in the size of the ectopic gestation from 24 mm to 16 mm, with a significant synchronous reduction of blood supply on the 8th day of MTX administration (Figure 4). The β-hCG fell gradually to non-pregnant levels within 36 days. After 6 months, hysteroscopy revealed a normal uterine cavity. A normal pregnancy occurred after 8 months, ending with a caesarean section for fetal distress.
Case report 2 A 37-year-old nulliparous woman, with 9 weeks’ amenorrhoea, presented to us complaining of vaginal spotting and acute abdominal pain. A transvaginal scan showed an eccentrically gestational sac of 29 mm, with embryonic viability, located at the outer right angle of fibromatous uterus. The pregnancy appeared implanted on the right extremity, where no myomas were present. The site of the gestational sac and the presence of the interstitial line sign suggested IP. The initial β-hCG value was 24, 213 mIU/l. MTX multidose protocol was administered as described above. On the 4th day, the β-hCG value was 16, 600 mIU/l, at the end of treatment on the 8th day 12, 1,312 mIU/l (Figure 5). Ultrasounds were performed on days 2, 5, 10 and then weekly until the risk of rupture, due to persistent trophoblastic proliferation, was presumed to disappear. This risk was evaluated by Doppler flow persistence in implantation site, which disappeared after 10 days from the beginning of treatment when the β-hCG level was 14.181 mIU/l. Ultrasound confirmed the resolution of the ectopic mass, 15 days after the termination of treatment.
Case report 3 A 33-year-old nulliparous woman with 7 weeks’ amenorrhoea, after a successful cycle of IVF, presented in our institution for vaginal bleeding. Ultrasonography revealed a singleton gestation, eccentrically placed in the left utero-tubal junctional area, lying 1 cm outside the most upper lateral edge of the decidualised cavity, measuring 0.4 cm. No embryo was visualised. Spectral tracing revealed a low resistant pattern deriving from trophoblastic tissue. The initial β-hCG was 1,351 mIU/l. Multidose protocol was administered. On the 8th day, β-hCG was 1,451 mIU/l, after 1 week 115 mIU/l and after another 5 days, the β-hCG level fell to 3 mIU/l.
Discussion The surrounding muscular wall of the interstitial portion confers ability to expand higher than other parts of fallopian tube.
