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Editorial

Volume 1 Issue 2, February 2014

Editorial
Gupta AS

Retrospective Diagnosis of Advanced Carcinoma Endometrium from Hepatic Nodule
Panchbudhe S, More V, Mali K, Satia MN.

 Cesarean Scar Ectopic Pregnancy
Khadkikar R, Wakchaure G, Prasad M, Chauhan AR.

 Heterotopic Pregnancy After IVF-ET
More V, Panchbudhe S, Mali K, Satia MN.

Robert’s Uterus
Channawar S, Chamariya S, Chauhan AR, Mayadeo NM.

 Pregnancy in a case of Constrictive pericarditis
Chakre S, Pardeshi S, Warke HS, Mayadeo NM.

Simultaneous Scar and Tubal Twin Ectopic Gestation
Jamdade K, Gupta AS.

 Conservative Management of Corpus Luteum Hematoma
Pandey N, Gupta AS.

 Hemoperitoneum In Coagulopathy: Conservative Treatment
Ansari MF, Parulekar SV.

Myomectomy: Lateral Extraperitoneal Approach
Parulekar SV

Editorial
Dr. A.S.Gupta
Co-Editor
Ectopic pregnancy is a major concern in reproductive age women. About 1 to 2 % of pregnancies are located in the extra uterine sites. It can disrupt the fertility potential of a young woman. Advances in assisted reproductive technologies (ART), super ovulation, better antimicrobial therapy of pelvic inflammatory disorders, conservative treatment modalities for ectopic gestations and increasing rate of cesarean deliveries have not only increased the incidence of ectopic gestations but pregnancy has found new sites to  implant. Further, twining in ectopic gestations has also increased. Simultaneous presence of two or more pregnancies in different sites is known as heterotopic pregnancy. Usually one pregnancy is intrauterine and the other is extrauterine situated frequently in the fallopian tube. Incidence of heterotopic gestations has increased from 1 in 30,000 to 1 in 3900 after the use of ART for conception. An incidence of 1.5 heterotopic pregnancies for every 1000 ART pregnancies has been reported in the United States during the period between 1999 and 2002. Pregnancies conceived with ART show an incidence of 11.7% of heterotopic pregnancy and reduction of tubal ectopic to 82.2% from  over 90% in spontaneous conceptions. This incidence is probably higher due to greater incidence of tubal disease, higher estrogen levels, and greater viscosity of the transfer medium and the method of embryo transfer. Heterotopic pregnancies pose diagnostic challenges as after detection of one pregnancy (usually an intrauterine one) an additional pregnancy is not considered as the symptoms and signs of early pregnancy, threatened abortion, like amenorrhea, pain in abdomen, nausea, vomiting and vaginal bleeding overlap. Significant abdominal pain with an intrauterine gestation should alert the clinician to the possibility of a simultaneous ectopic gestation. β HCG levels in a heterotopic pregnancy usually reflect the intrauterine rather than the ectopic gestation. The ectopic pregnancy usually gets diagnosed only after a catastrophic event like a hemoperitoneum occurs. Such eventualities can be averted provided all clinicians are alert and vigilant and they think ‘ectopic’ in every woman who presents with an early pregnancy. Exploratory laparotomy is needed if hemoperitoneum is suspected. However, laparoscopy aids in the diagnosis in a stable patient. Treatment needs to be personalized to the ectopic site. Live intrauterine pregnancy and a coexisting unruptured ectopic gestation cannot be managed conservatively by systemic methotrexate. Conservative laparoscopic surgery or ultrasonic local injection of potassium chloride in the gestational sac can be alternative treatment modalities in a stable patient.
Cesarean Scar Ectopic Gestation: With the increasing cesarean births complications in the subsequent pregnancies like morbid adhesions of placenta, placenta previa and scar pregnancies are on the rise. These are type of intramural or an intramyometrial pregnancy. The pregnancy implants on the previous cesarean or hysterotomy scar and is surrounded all round by the myometrium and is overtly unconnected with the endometrial cavity. Some of these pregnancies are partially in the uterine cavity and grow till term. Pregnancies that have completely invaded the myometrium tend to rupture in the 1st trimester. Microscopic or wedge defects in the scar allow the pregnancy to burrow in the myometrium and implant there. Besides cesarean and hysterotomy scars, false tracts due to intrauterine device and previous dilatation and curettage, in vitro fertilization and adenomyosis are some other risk factors for scar pregnancies. Various ultrasonographic diagnostic features like bulging of the cesarean scar especially in close proximity of the bladder, bulge or distortion of the anterior outline of the uterus, and a gestation sac with a live embryo have been reported. Doppler signals may further aid in the diagnosis. Close differential diagnosis will be a cervical pregnancy and morbid adhesion of the placenta. Surgical excision of the pregnancy and the scar with repair is the preferred treatment modality even in stable patients as it not only allows the resection of the pregnancy but also allows the clinician to excise and repair the scar. Conservative treatments like systemic methotrexate, local ultrasonographic administration of injection of potassium chloride in the sac are the treatment options for an unruptured scar pregnancy. However, these patients need prolonged follow up as the resolution of the pregnancy is very slow and the chance of uterine rupture and hemorrhagic shock remain. Hysterectomy and uterine artery embolization have also been used in select patients. In subsequent pregnancies these patients are at an increased risk for re-implantation at the scar, placenta accreta, scar rupture and severe hemorrhage. An early ultrasound in the next pregnancy is recommended to see for the site of implantation.
A patient with a heterotopic gestation presented in this issue had one of the rarer types of twin ectopic gestations. Both the pregnancies were extra uterine in location. This patient was extremely fortunate. The preoperative diagnosis of ruptured tubal ectopic and a normal intrauterine pregnancy was proven wrong intraoperatively. An unruptured tubal ectopic and a ruptured scar ectopic was seen on exploratory laparotomy. Had it been the other way round tubal rupture would have been treated and the scar ectopic would have been missed. It would then have subsequently presented with a catastrophe and probably a delayed diagnosis and its consequences. In another case, the patient had an intrauterine and a cervical ectopic pregnancy. In the near future the scientific community is likely to encounter more of such cases. Awareness and the various modes of presentation are aids to an early diagnosis of this condition. Treatment should be determined by the site of implantation and the condition of the patient.
It gives me great pleasure to present this second issue of the journal to the readers and hope the cases in this will give insight into various aspects in clinical practice.

Retrospective Diagnosis of Advanced Carcinoma Endometrium from Hepatic Nodule



Author Information
Panchbudhe Shruti*, More Vibha**, Mali Kimaya***, Satia MN****.
(* Assistant Professor, ** Assistant Professor, *** Assistant Professor, **** Professor Department of Obstetrics and Gynecology, Seth GS Medical College & KEM Hospital, Mumbai, India)

Abstract

We report an unusual case of a 34 year old, infertile, woman who presented in surgery outpatient department with abdominal distension and right shoulder pain. On clinical examination there was suspicion of subacute intestinal obstruction, and on evaluation with X-ray and ultrasonography, obstruction was ruled out. An incidental finding of liver nodule was noted on ultrasonography (USG) which was further confirmed on computerized tomography (CT). CT guided biopsy of liver nodule was suggestive of endometrioid carcinoma. Patient was referred to gynaecological department to rule out primary focus and was diagnosed retrospectively to have carcinoma endometrium on fractional curettage. This is an unusual presentation of advanced carcinoma endometrium in the third decade of life which in a normal situation is the malignancy of fifth and sixth decades of life with the only risk factor of nulliparity without any gynecological symptoms.

Introduction

Endometrial carcinoma has four histological subtypes of which the endometrioid subtype is the most common, affecting 75–80% of patients.  Endometrioid endometrial adenocarcinoma has a better prognosis than the other subtypes (papillary serous, clear cell or carcinosarcoma) and can metastasize to the lung, bone, brain or liver as solitary deposits amenable to resection.[1] Liver metastases from endometrial cancer at the time of presentation represent true haematogenous spread. Histopathology of typical endometrioid adenocarcinoma is usually characterize by back to back arrangement of endometrial-type of glands of varying differentiation/atypia with no intervening stroma. . Endometrioid adenocarcinoma have three grades on histopathology. Grade 1 consists of 5% or less of nonsquamous or nonmorular solid growth (well differentiated), Grade 2 consists of 6% to 50% of solid growth
( moderately differentiated) , and Grade 3 consists of more than 50% of solid growth (poorly differentiated).The various variants of endometrioid adenocarcinoma includes villoglandular, secretory, ciliated cell, and adenocarcinoma with squamous differentiation type of pattern, each having a peculiar histopathology along with the usual features of typical endometrioid adenocarcinoma.[1]

Case Report

A 34 year old nulliparous woman, married for 14 years, presented to the surgery outpatient department with complaints of chronic right sided shoulder pain for 1 year and abdominal distension for last 6 months.On general examination  she was averagely built and nourished. Her vital parameters were stable. She attained menarche at the age of 13 years and her past and present menstrual cycles were regular with average amount of flow. Abdominal examination revealed distension, moderate ascites, minimal guarding and no rigidity. A clinical impression of subacute intestinal obstruction was made. However plain radiograph of the abdomen AP view and USG ruled out intestinal obstruction. USG showed a cystic lesion in right lobe of liver with moderate ascites.  CT of abdomen also showed a heterogeneous mass about 3X3 cm in the right lobe of liver with ascites. Ascitic fluid tapping was done. It did not show any malignant cells. CT guided liver biopsy was done and tissue was sent for immunohistochemisty and histopathology. Immunohistochemistry revealed expression of Cdx2 protein.  Histopathological examination of the tissue showed small fragments composed of different glands with squamous differentiation. Hence a diagnosis of well differentiated endometrioid neoplasm was made and the patient was then referred to gynecology department to rule out primary focus of malignancy. On gynecological evaluation the patient was a case of primary infertility with history of diagnostic hysterolaparoscopy done 6 years ago. She had taken treatment for infertility, details of which were not available. Her menstrual cycles were normal. She was non diabetic and non hypertensive. On abdominal examination an infraumbilical scar of laparoscopy was present. There was moderate ascites. On speculum examination cervix and vagina were healthy and on bimanual examination the uterus was anteverted, bulky, soft, mobile and bilateral fornices were free. Rectal examination showed absence of any nodularity and free parametrium. Pap smear revealed chronic inflammation with few dysplastic cells. Ultrasonography of pelvis revealed uterus 10×7×6 cm with thick polypoidal endometrium of 2cm. Chest radiograph was normal. Other routine preoperative investigations were normal. A fractional curettage was performed. Histopathological report of the endometrial tissue was suggestive of well differentiated endometrioid adenocarcinoma of the endometrium. Hence the diagnosis of stage IV B carcinoma endometrium with hepatic metastasis was made. In view of advanced carcinoma endometrium the patient was referred to an oncology center for further treatment. She was advised cytoreductive surgery followed by adjuvant radiotherapy. But patient did not follow up and died after 8 months from diagnosis of advanced carcinoma endometrium.


Figure 1. Histopathological appearance of the tumor. A. Low power; B. High power.
Discussion

Endometrial cancer is a disease that occurs primarily in postmenopausal women in the sixth and seventh decades of life and is virulent with advancing age. The risk for endometrial cancer increases with unopposed estrogen. Various conditions associated with an increased risk of endometrial carcinoma include obesity, nulliparity, early menarche, and late menopause. Obesity appears to pose the greatest risk due to aromatization of androstenedione to estrone in peripheral fat. Diabetes mellitus, hypertension, high fat diet and family history of endometrial, colon, ovarian cancer have also been implicated as the  additional risk factors for endometrial carcinoma. Infertility and a history of irregular menses as a result of anovulatory cycles are also documented risk factors for developing endometrial cancer.[2] Women with endometrial carcinoma have vaginal bleeding or discharge as the only presenting symptom in 90% of the cases and less than 5% of the women are asymptomatic. Sometimes women may experience pelvic pressure or discomfort due to uterine enlargement or extrauterine spread of the disease and rarely some may present with haematometra or pyometra due to cervical stenosis which is a poor prognostic indicator of endometrial carcinoma.[3]. In contrast to the cervix, where cytological screening has been enormously successful in preventing cervical cancer, there is no effective general population screening of asymptomatic women for endometrial cancer. Endometrial carcinoma is usually detected in asymptomatic women due to abnormal Pap test, histopathology of the uterine specimen removed for some other indication or abnormal finding on abdominal and pelvic USG or CT scan obtained for an unrelated reason as it was seen in our case. Those women who have malignant cells on Pap test are likely to have a more advanced stage of disease.[4] Physical examination does not have any positive finding, although obesity and hypertension are commonly associated constitutional factors. Abdominal examination does not show any abnormality, except in advanced cases in which ascites or hepatic or omental metastases may be palpable. A detailed gynaecological examination should be performed which includes inspection of vagina and cervix along with bimanual rectovaginal examination to evaluate the uterus for size and mobility, the adnexa for masses, the parametria for induration, and the cul-de-sac for nodularity. All patients with endometrial cancer should undergo surgical staging which includes selective pelvic and para-aortic lymph node dissection, in addition to hysterectomy and salpingo-oophorectomy, based on the intraoperative assessment of risk for lymph node metastasis or other extrauterine spread. Surgical staging identifies most patients with extrauterine disease and has a significant impact on treatment decision. Stage IV endometrial adenocarcinoma, in which tumor invades the bladder or rectum or extends outside the pelvis, makes up about 3% of cases.[5,6,7] Several reports have noted a positive impact of cytoreductive surgery on survival, the median survival being about 3 times greater with optimal cytoreduction (18–34 months versus 8-11 months, respectively).[8,9] In our case due to noncompliance of patient her survival was reduced to 8 months. Treatment for stage IVB endometrial cancer involves one of tumor-reduction or palliative chemotherapy or radiation. Tumor-reductive surgery is typically followed with adjuvant chemotherapy, hormonal therapy, and/or radiation therapy although radiotherapy is the treatment of choice. Resection of liver capsule metastases as part of cytoreductive surgery has been shown to be feasible and may prolong survival.[10] Palliative radiotherapy and platinum-based chemotherapy regimens followed by progestogens can also be considered in patients with endometrial carcinoma with liver metastasis which is effective in both preventing recurrence and prolonging survival. Patients with suspected advanced stage endometrial carcinoma should be counseled on the potential benefits of optimal cytoreductive surgery and alternative treatment options should be considered in those  with surgically nonresectable disease.

References

  1. Fanning J, Evans MC, Peter AJ, et al. Endometrial adenocarcinoma histological subtypes; clinical and pathological profile. Gynecol Oncolm1989;32:288-291.
  2. Brinton LA, Berman ML, Mortel R, et al. Reproductive, menstrual and medical risk factors; for endometrial cancer: results from a case control study. Am J Obstet Gynecol 1993;81:265-271.
  3. Smith M, McCartney AJ. Occult, high-risk endometrial carcinoma. Gynecol Oncol 1985;22:154-161.
  4.  Dubeshter B, Warshal DP, Angel C, et al. Endometrial carcinoma: the relevance of cervical cytology. Obstet Gynecol 1991;77:458-462.
  5. Pliskow S, Penalver M, Averette HE. Stage III and IV endometrial carcinoma: a review of 41 cases. Gynecol Oncol 1990;38:210-215.
  6. Aalders JG, Abeler V, Kolstad P. Stage IV endometrial carcinoma: a clinical and histopathological study of 83 patients. Gynecol Oncol 1984;17:75-84.
  7. Goff BA, Goodman A, Muntz HG, et al. Surgical stage IV endometrial carcinoma: a study of 47 cases. Gynecol Oncol 1994;52:237-24.
  8. Bristow RE, Zerbe MJ, Rosenshein NB, et al. Stage IV endometrial carcinoma: the role of cytoreductive surgery and determinants of survival. Gynecol Oncol 2000;78:85-91.
  9. Chi DS, Welshinger M, Venkatraman ES, et al. The role of surgical cytoreduction in stage IV endometrial carcinoma. Gynecol Oncol 1997;67:56-60.
  10. Tangjitgamol S, Levenback CF, Beller U, Kavanagh JJ. Role of surgical resection for lung, liver, and central nervous system metastases in patients with gynaecological cancer: a literature review. Int J Gynecol Cancer.2004;14:399–422.

Citation

Panchbudhe S, More V, Mali K, Satia MN. Retrospective diagnosis of advanced Carcinoma Endometrium from hepatic nodule. JPGO 2014 Volume 1 Number 2 Available from: http://www.jpgo.org/2014/02/retrospective-diagnosis-of-advanced.html

Cesarean Scar Ectopic Pregnancy

Author Information
Khadkikar Rashmi*, Wakchaure Ganesh**, Prasad Madhva***, Chauhan AR****
(* Assistant Professor, ** Assistant Professor, *** Third Year Resident, **** Additional Professor, Department of Obstetrics and Gynecology, Seth G.S. Medical College and K.E.M Hospital, Mumbai, India.)

Abstract

Cesarean scar ectopic is one of the rarest forms of ectopic pregnancy where the gestational sac is implanted in the myometrium at the site of previous cesarean section (CS) scar.  Transvaginal ultrasonography (USG) and Doppler are diagnostic. Complications such as uterine rupture and hypovolemic shock may be life-threatening and hamper fertility; if diagnosed early, conservative management with systemic or local methotrexate, or uterine artery embolization (UAE) to prevent hemorrhage, is possible if the patient is hemodynamically stable. We report a case of cesarean scar ectopic managed conservatively with methotrexate therapy.

Introduction

The first case of cesarean scar ectopic was reported in 1978.[1]  Incidence varies from 1:1800[2] to 1:2226[3] of all pregnancies, with increasing incidence due to increased number of both primary and repeat CS.[4] Associated risk factors are previous CS, and procedures which cause endometrial damage like myomectomy, adenomyosis, IVF and embryo transfer, dilation and curettage (D&C), and manual removal of placenta. It has been proposed that pregnancy enters the cesarean scar via microscopic fistulae.[4]

Case Report

A 27 years old second gravida with one previous LSCS was admitted to the emergency room of our tertiary institute with complaints of painless vaginal bleeding since 2 to 3 weeks. Bleeding was sudden in onset, without prior amenorrhea, with spotting on some days, alternating with episodes of profuse bleeding. On examination, pallor and tachycardia were noted. Per abdominal examination was normal. On vaginal examination, uterus was anteverted, bulky, fornices were unremarkable. Urine pregnancy test was positive.
Ultrasonography showed normal-sized uterus with a 0.5cm cystic area with surrounding hyper-echogenecity at the LSCS scar site, suggestive of a gestational sac. Doppler showed. There was no evidence of gestational sac in endometrial cavity. Minimal free fluid was seen in cul-de-sac.

Figure 1: Transvaginal USG showing normal endometrial cavity and gestational sac in LSCS scar.

Figure 2: Doppler showing high velocity, low impedance peri-trophoblastic flow.

As patient was hemodynamically stable, sac size was small and initial serum βhCG was 6484mIU/ml, decision for medical treatment with methotrexate (40mg IM at 1mg/kg body weight) was taken after baseline biochemistry. However, βhCG remained the same after the first dose of methotrexate; hence transvaginal USG-guided intragestational injection of 40mg methotrexate was performed, followed by leucovorin rescue (0.4mg IM). Subsequently, βhCG fell to 3009mIU/ml after 1 week (>50% decrease), 1026mIU/ml after 2 weeks and 100mIU/ml after 6 weeks. Two weeks after the procedure, sonography showed no lesion or vascularity.

Discussion

Patients of scar ectopic commonly present with painless, sometimes heavy, vaginal bleeding. Uterine tenderness may be elicited if the ectopic is in process of rupture. Unlike adherent placenta where the placenta invades the myometrium at the scar site but the pregnancy is uneventful, scar ectopic is more aggressive in its behaviour because of its early invasion which can lead to rupture, and massive hemorrhage.

Diagnosis is made by USG and Doppler; MRI is confirmatory. Diagnostic criteria on Doppler to differentiate from cervical ectopic pregnancy are:[3] 
  1. Gestational sac located between bladder wall and anterior isthmic portion of uterus
  2. No trophoblastic tissue in uterine cavity or cervical canal
  3. Clearly visible circular blood flow surrounding the sac
Jurkovic has described a negative “sliding organ sign” as diagnostic of scar ectopic –the inability to displace the gestational sac from its position at the level of the internal os by gentle pressure applied by the transabdominal probe.[4]

Mode of treatment depends on the presentation as rupture and severe hemorrhage may warrant hysterectomy. Cesarean scar pregnancy may be managed conservatively if diagnosed early. Conservative modalities include excision of trophoblastic tissue through hysterolaparoscopy; intralesional KCl, local or systemic methotrexate; D&C under laparoscopic guidance with Foley balloon tamponade.  However, D&C should not be done as first line treatment due to risk of perforation and catastrophic hemorrhage.[5] Selective UAE may be prophylactically used with any conservative procedure to prevent excessive bleeding and conserve fertility.

Intragestational injection of methotrexate is preferable to systemic regimen as it achieves a high concentration locally and interrupts the pregnancy more rapidly. Baseline βhCG of >5,000mIU/ml is associated with failure of systemic methotrexate and better response to intralesional administration5. Success of treatment is monitored by hemodynamic status, regression of gestational sac on USG, and falling βhCG levels, as seen in our case.

In a hemodynamically stable patient, laparoscopy is the best choice as it provides both diagnosis and definitive management. Surgical technique described is vasopressin injection in the tissue surrounding the sac to reduce bleeding, excision and retrieval of the scar ectopic mass, and closure of the defect and hemostasis with endosutures and bipolar cautery.[6]
Long-term outcomes after conservative treatment include concerns about future fertility and recurrence of cesarean scar ectopic. As uterine rupture can occur in the next pregnancy, the scar should be evaluated hysteroscopically before next pregnancy and by USG during subsequent pregnancies.[5]
To conclude, for women with cesarean scar ectopic who desire fertility, in the absence of hemodynamic instability, conservative treatment with systemic or local methotrexate is an excellent option.

References
  1.  Larsen JV, Solomon MH. Pregnancy in a uterine scar sacculus: an unusual cause of postabortal haemorrhage. S Afr Med J 1978;53:142-3.
  2. 2.      Seow KM, Huang LW, Lin YH, Yan-Sheng Lin M, Tsai YL, Hwang JL. Caesarean scar pregnancy: issues in management. Ultrasound Obstet Gynecol 2004; 23:247-53.
  3. Ash A, Smith A, Maxwell D. Caesarean scar pregnancy. BJOG 2007;114:253-263.
  4.  Jurkovic D, Hillaby K, Woelfer B, Lawrence A, Salim R, Elson CJ. First trimester diagnosis and management of pregnancies implanted into the lower uterine Caesarean section scar. Ultrasound Obstet Gynecol 2003;21:220-7.
  5. Rotas MA, Haberman S, Levgur M. Cesarean scar ectopic pregnancies: etiology, diagnosis and management. Obstet Gynecol 2006;107:1373-7.
  6. Wang YL, Su TH, Chen HS. Operative laparoscopy for unruptured ectopic pregnancy in a caesarean scar. BJOG 2006;113:1035-8.
Citation

Khadkikar R, Wakchaure G, Prasad M, Chauhan AR. Cesarean Scar Ectopic Pregnancy. JPGO Volume 1 Issue 2, February 2014, available at:http://www.jpgo.org/2014/02/cesarean-scar-ectopic-pregnancy.html

Heterotopic Pregnancy After IVF-ET



Author Information
More Vibha*, Panchbudhe Shruti**, Mali Kimaya***, Satia MN****
(* Assistant Professor, ** Assistant Professor, *** Assistant Professor, **** Professor
Department of Obstetrics and Gynecology, Seth G.S. Medical College and K.E.M Hospital, Mumbai, India.)

Abstract

We report a case of heterotopic pregnancy, cervical-intrauterine, conceived with in-vitro fertilization. She was successfully managed with conservative systemic Methotrexate therapy.

Introduction

Heterotopic pregnancy is coexistence of intra- and extrauterine pregnancy and is rare. Cervical- intrauterine is a rare variety of heterotopic pregnancy. Cervical pregnancy is a rare and life-threatening form of ectopic pregnancy. Improved sonographic techniques have allowed earlier diagnosis and successful attempts at conservative management.

Case report

A 43 year old woman, married for 12 years, gravida 2, abortion 1, with 7 weeks of gestation was referred to us in view of non-viable cervical ectopic and intrauterine pregnancy after in-vitro fertilization and embryo transfer. She had no complaints. Findings of diagnostic hysteroscopy and laproscopy done 10 years ago were normal. Findings with bilateral tubes patent and was started on treatment. She had conceived once with ovulation induction and intrauterine insemination. She developed a missed abortion for which dilatation and curettage was done. She underwent hysteroscopy and cervical dilatation five years later. Hysteroscopic findings were normal. Endometrial TB-PCR was positive for mycobacterium tuberculosis complex. She was treated with antitubercular therapy for 6 months. She then underwent ovarian hyperstimulation with long protocol, 6 embryo were retrieved of which 4 embryos were transferred. She conceived in the same cycle. She was started on progesterone support and low dose aspirin. Serum β-hCG level was 593 mIU/mL 15 days after the embryo transfer. After 6 weeks ultrasonography was done. It showed non viable twin gestation. Gestational sac 1 corresponding to 6.4 weeks was low lying in the uterus and gestational sac 2 corresponding to 5.4 weeks was located in the cervix. Then she was referred to our institute for further management.

Figure 1: Ultrasonic scan showing intrauterine (solid arrow) and cervical (hollow arrow) pregnancy.

On examination her vital parameters were stable. Systemic examination showed no abnormality. Her abdomen was soft and non tender. On per speculum examination the external os was closed, the cervix was slightly enlarged. There was no bleeding. Bimanual pelvic examination was deferred so as not to disturb the cervical ectopic pregnancy. Serum β-hCG levels on admission was 54,112 mIU/ml. Hemogram , hepatic function and renal function tests were within normal limits.
Conservative management with Methotrexate administration was undertaken in an attempt to preserve her fertility. The regimen consisted of intramuscular Methotrexate 50 mg (1 mg/kg) on day 1, 3 and 5 and Folinic acid 5mg (0.1 mg/kg) on day 2, 4 and 6. The patient was monitored with serial β-hCG and ultrasound with Doppler flow.
 Serial β-hCG levels
Day (since hospitalization)
Serum β-hCG levels (mIU/ml)
1
54,112
3
37,635
5
28,759
7
15,557
15
578
22
79
37
< 2 mIU/ml

Ultrasonography showed a decrease in size of both gestational sacs and Doppler showed decreased peak systolic velocity. She was monitored for Methotrexate toxicity with complete blood count and hepatic function test. She was then discharged on day 15 with advised to follow-up with β-hCG level after one week or if she had any complaints like bleeding per vaginum or pain in abdomen. Subsequent menstrual periods were normal.

Discussion

Heterotopic pregnancy is coexistence of intra- and extrauterine pregnancy. The prevalence is low in spontaneous pregnancy(1 in 30,000).[1] With the use of ovulation induction and assisted reproductive techniques; the incidence of heterotopic pregnancy has risen up to 0.75%-1.5%.[1]  As opposed to ectopic pregnancy which are diagnosed and treated at preclinical stage. Heterotopic pregnancies are often diagnosed after clinical symptoms and signs develop. Cervical- intrauterine is a rare variety of heterotopic pregnancy. Cervical ectopic pregnancy is uncommon and life threatening. The incidence reported occurring in 1 of 8628 deliveries.[2]  Its incidence is increasing as a result of in-vitro fertilization.[1]. The other predisposing factors include  cervical dilatation and curettage and previous cesarean section.
Cervical ectopic pregnancy is frequently confused with neoplastic growth or spontaneous abortion. Ultrasonographic diagnosis is useful in the early detection of cervical pregnancy. Ultrasonographic findings include the hourglass uterus or dilated cervix. Doppler flow sonography is helpful only in distinguishing abortions in progress from those with vascular implantation in the cervix.
Management of cervical ectopic is surgical or medical. Surgical management includes dilatation and curettage, in early cervical pregnancy or hysterectomy in uncontrolled, life-threatening bleeding,. Preoperative preparation  prior to dilatation and curettage to reduce vascularity  includes transvaginal ligation of cervical branch of uterine artery, Shirodkar cerclage, uterine artery embolization, or intracervical vasopressin injection. To control post evacuation bleeding, Foley catheter with a 30 ml balloon is inflated.  Medical therapy can be used as primary treatment for cervical pregnancy or adjunct to surgical treatment. It includes systemic Methotexate alone, systemic Methotexate with local  (intraamniotic or intracervical) injection of either Methotexate or potassium chloride.

References

1. Mark A. Damario,  John A. Rock. Ectopic Pregnancy. Te Linde’s Operative Gynaecology 10th ed. New Delhi: Wolters Kluwer Health- Lippincott Williams & Wilkins 2008;pp. 819-821
2. Ushakov FB, Elchalal U, Aceman PJ, Schenker JG. Cervical pregnancy: past and future. Obstet Gynecol Surv 1997;52: 45–59.
3. Weyerman PC, Verhoeven ATM, Alberda ATM. Cervical pregnancy after in vitro fertilization and embryo transfer. Am J Obstet Gynecol 1989;161:1145–1146.

Citation

More V, Panchbudhe S, Mali K, Satia MN. Heterotopic Pregnancy After IVF-ET. JPGO Volume 1 Issue 2, February 2014, available at :http://www.jpgo.org/2014/02/heterotopic-pregnancy-after-ivf-et.html

Robert’s Uterus



Author Information
Channawar Sarita*, Chamariya Sumit**, Chauhan AR***, Mayadeo NM****
(* Assistant Professor, ** Third Year Resident, *** Additional Professor, **** Professor
Department of Obstetrics and Gynecology, Seth G.S. Medical College and K.E.M Hospital, Mumbai, India.)

Abstract

Developmental anomalies of the mullerian duct system represent some of the most fascinating and challenging disorders that gynecologists encounter. This is a report of a normally menstruating adolescent girl who presented with acute abdominal pain aggravated during menses. On ultrasonography, she had a septate uterus with non-communicating unilateral (right-sided) hematometra, a very rare müllerian anomaly termed as Robert’s uterus; diagnosis was confirmed on magnetic resonance imaging (MRI). The patient underwent laparotomy with complete excision of the non-communicating hemiuterus and endometrial cavity with drainage of hematometra; she recovered completely and resumed painless menses. The case and relevant literature review are presented.

Introduction

The reported incidence of mullerian duct anomalies (MDAs) is 0.1-3.5%.[1] The most commonly reported anomalies are septate, arcuate, didelphys, unicornuate, or hypoplastic uteri. The widely accepted American Fertility Society (AFS) classification organizes MDAs according to major uterine anatomic defect and allows for standardized reporting methods. Septate uterus (AFS Class V) is the most common, resulting from incomplete resorption of the medial septum after complete fusion of the mullerian ducts has occurred; variations exist like complete, partial and segmental. The complete septum extends from the fundal area to internal os and divides the endometrial cavity into 2 components and is often associated with a longitudinal vaginal septum. The most common presenting symptoms are dysmenorrhea, dyspareunia, primary or secondary infertility, pregnancy loss and obstetric complications.

Case Report

A 20 years old unmarried girl, normally menstruating for 5 years, presented to emergency room with severe dysmenorrhea. On examination, her secondary sexual characters were normal. She had mild pallor, marked lower abdominal tenderness but no palpable lump, and normal external genitalia. On rectal examination, a bulky uterus was felt in midline, deviated to the right. Transabdominal and transperineal ultrasonography revealed a septate uterus with right sided hematometra with normal left endometrial cavity. MRI confirmed the diagnosis and clearly showed septate uterus with normal left-sided outflow tract with blind right cavity with septum till internal os and single cervix. There was no communication between the two cavities; there was no evidence of any renal anomaly. The noncommunicating horn was considered to be the cause of the patient's severe dysmenorrhea due to intracavitary retention of menstrual effluent.

Figure 1: MRI showing Robert’s uterus with right-sided hematometra.

The patient underwent exploratory laparotomy. The uterus was bulky with normal fundal contour and well-developed left side, with a soft, bulging right side suggestive of hematometra. Though right adnexae were normal in appearance, the fallopian tube was not communicating with the right hemiuterus. An incision was taken over the right hemiuterus; anchovy sauce-like material of approximately 100 ml was drained. As the septum was reaching only till internal os, there was neither communication between lower extent of right hemiuterus and cervix below, nor with left-side cavity. Hence non-communicating blind right hemiuterus was excised completely with endometriectomy. The right round ligament was divided; the bladder pushed inferiorly, extent of incision was up to the internal os inferiorly and lateral to the midline septum, so as to preserve the normal functioning left hemiuterus. Intraoperative findings were consistent with preoperative diagnosis. Uterine incision was sutured in two layers using delayed absorbable sutures.

Figure 2: Intraoperative findings

Figure 3: Excision of right hemiuterus

Figure 4: Uterus after excision of right hemiuterus and closure

Histopathological examination was consistent with hematometra. The patient’s postoperative recovery was uneventful with resumption of normal painless menses in the next cycle. 

Discussion

Robert’s uterus or asymmetric septate uterus is a rare variant of septate uterus, a unique congenital mullerian anomaly first reported by Robert in 1970[2]; so far very few cases have been reported in literature.[3,4,5] This is characterized by a complete septum, non-communicating hemiuteri with one blind horn causing hematometra and one communicating hemiuterus with single cervix and a normal extrauterine morphology. Patients usually present in postmenarcheal period with unilateral hematometra causing dysmenorrhea. The modalities for diagnosis are ultrasonography and MRI while hysterosalpingography, hysteroscopy, and laparoscopy are useful especially in infertility cases. MRI provides excellent tissue characterization helping in reliably differentiating septate from bicornuate uterus and also in diagnosing asymmetric septate uterus.[6,7]
Surgery can be done by open or minimally invasive method, definitive surgery involves drainage of hematometra and excision of blind non-communicating hemiuterus taking care to maintain integrity of functional communicating hemiuterus and cervix.[8, 9]
In order to avoid inappropriate management, gynecologists should be aware of this rare entity while evaluating cases of severe dysmenorrhea in previously normal menstruating young girls. Prompt early diagnosis and surgical correction are essential to avoid future morbidity due to endometriosis. Few cases have reported successful pregnancy after endometriectomy in these cases.[10]

References
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  1. Acién P. Incidence of Mullerian defects in fertile and infertile women. Hum Reprod 1997;12(7):1372-6.
  2. Gupta N, Mittal S, Misra R, A unique congenital mullerian anomaly Robert's uterus. Arch Gynaecol Obstet 2007 Dec;276(6):641-3.
  3. Capito C, Sarnacki S. Menstrual retention in a Robert's uterus, J Pediatr Adolesc Gynecol 2009 Oct; 22(5):e104-6.
  4. Benzineb N, Bellasfar M, Merchaoui J, Sfar R. Robert's uterus with menstrual retention in the blind cavity. J Gynaecol Obstet Biol Reprod (Paris),1993;22(4):366-8.
  5.  Rebelo T, Almeida e Sousa LA, Sampaio MG, Martins MJ, et al. Asymmetric septate uterus with unilateral menstrual retention a  rare uterine malformation, Acta Med Port, 1997;10(10):721-4.
  6. Marcal L, Nothaft MA, Coelho F, Volpato R, et al. Mullerian duct anomalies: MR imaging. Abdom Imaging 2011; 36(6):756-64.
  7. Bermejo C, Martínez Ten P, Cantarero R, Diaz D, Pérez Pedregosa J, Barrón E,  Three-dimensional ultrasound in the diagnosis of Mullerian duct anomalies and concordance with magnetic resonance imaging. Ultrasound Obstet Gynecol 2010; 35(5):593-601.
  8. Takeuchi H, Sato Y, Shimanuki H, Kikuchi I, Kumakiri J, Kitade M, Kinoshita K, Accurate preoperative diagnosis and laparoscopic removal of the cavitated non-communicated uterine horn for obstructive Mullerian anomalies. J Obstet Gynaecol Res 2006;32(1):74-9.
  9. Perino A, Chianchiano N, Simonaro C, Cittadini E, Endoscopic management of a case of complete septate uterus with unilateral hematometra, Human Reprod 1995;10(8):2171-3.
  10. Vural M, Yildiz S, Cece H, Camuzcuoglu H. Favourable pregnancy outcome after endometrectomy for a Robert's uterus. J Obstet Gynaecol 2011;31(7):668-9.
Citation
Channawar S, Chamariya S, Chauhan AR, Mayadeo NM.  Robert’s Uterus. JPGO 2014 Volume 1 Number 2 Available from:http://www.jpgo.org/2014/02/roberts-uterus.html