Cervical ectopic pregnancy at 22 weeks: A case with subsequent pregnancy outcome

The patient provided consent for this case report, including the sharing of de‐identified radiology images.

The authors received no specific funding for this work.

Fertility‐sparing management of CEP can avoid the morbidity of a gravid hysterectomy and allow patients to achieve family‐building goals. While first‐trimester conservative management of CEP is reported [ 2 ], second‐trimester CEP is rare. Our case contributes to the scant literature on second‐trimester fertility‐sparing CEP management with subsequent successful obstetric outcome. A reported successful uterine‐sparing management at 15 weeks had similarly been sonographically misdiagnosed with threatened abortion at 7 weeks. That pregnancy was managed with feticidal injection, systemic methotrexate, and removal of persistent fetal tissue in the cervix 8 weeks later; she eventually had a normal pregnancy and live birth [ 3 ]. Another case of a CEP at 23 weeks with live birth has been described [ 4 ]. Conversely, there are case reports of CEPs at 16 and 18 weeks resulting in hysterectomy. Tariq reported a patient with an acute abdomen who was diagnosed with a ruptured CEP upon laparotomy [ 5 ]. Coulter‐Nile reported a patient with previable rupture of membranes who failed induction and was diagnosed with CEP intraoperatively [ 6 ]. Jaswal [ 7 ] reported a patient at 18 weeks in hemorrhagic shock with products of conception in the cervix, resulting in maternal death. Pathology confirmed CEP. Our case and the case by Verma [ 3 ] demonstrate the limitations of early CEP diagnosis. Both patients had first‐trimester ultrasounds that reported threatened abortion without mention of possible CEP, delaying diagnosis until the second trimester. One study of 27 patients referred for suspected first‐trimester CEP reported 22% were ultimately diagnosed with CEP and 22% with threatened abortion [ 8 ], underscoring the difficulty in distinguishing between similar ultrasound appearances. Those authors suggested strong consideration for CEP with the following sonographic findings: gestational sac or placenta in the cervical canal, absence of intrauterine pregnancy with a visualized endometrial stripe, hourglass shape of uterus with a ballooned cervix, and positive embryonic/fetal cardiac activity. Persistent blood flow to the gestational sac is another proposed criterion. There is an absence of data regarding specificity of first trimester MRI for CEP diagnosis and broad use is limited by access and cost [ 8 ]. Our patient presented to our care after 20 weeks. Therefore, we relied on two imaging modalities in the second trimester to identify cervical extension of the placenta, short cervix with funneling, and placental invasion into the lower uterus and cervical canal. Together with the obtained images from her 9‐week ultrasound, her presentation was felt to be consistent with CEP; initial MRI showed minimal myometrium with mostly cervix surrounding the placenta. While abnormal placentation is inherent in CEP, our diagnosis of PAS was based on clinical findings at the time of surgery rather than histopathologic confirmation given the uterine‐sparing technique. Our patient underwent planned multimodal management involving intraamniotic digoxin, UAE, ureteral stent placement with hysterotomy, and balloon tamponade. Fowler et al. [ 2 ] reported that combination approaches for first‐trimester CEPs were associated with decreased odds of subsequent procedures compared to methotrexate alone. For CEPs at more advanced gestational ages, combination treatment involving a surgical approach is likely necessary given more advanced pregnancy development. Our patient demonstrated resolution of the retained cervical products by 5 months postpartum. Suppression of menses until resolution of cervical abnormalities was important to prevent pregnancy and avoid confounding of bleeding symptoms attributable to failed conservative management. While she was able to spontaneously conceive and carry a healthy pregnancy to term, it is unclear whether the postpartum hemorrhage secondary to uterine atony at the time of scheduled cesarean delivery was related to her prior PAS or CEP. We recommend maternal fetal medicine consultation for subsequent pregnancies after CEP with attention to pregnancy location and screening for cervical insufficiency.

Cervical ectopic pregnancies (CEPs) comprise <0.1% of ectopic pregnancies [ 1 ]. There is no standard management guideline. Medical treatments include systemic methotrexate, balloon catheter, intra‐amniotic methotrexate or potassium chloride, or uterine artery embolization (UAE). Surgical options include dilation & curettage (D&C) or hysterectomy, with the latter for uncontrollable hemorrhage or undesired fertility. Much of our understanding is drawn from case reports or series [ 2 ]. For CEPs diagnosed in the second trimester, the likelihood of successful fertility‐sparing treatments decreases. We report a case of CEP with placenta accreta spectrum (PAS) managed without hysterectomy at 22 weeks’ gestation, with subsequent successful pregnancy outcome. A 28‐year‐old healthy gravida 1 presented to the emergency department for first‐trimester bleeding in a spontaneously conceived pregnancy. Transvaginal ultrasound demonstrated a viable 9‐week singleton pregnancy. The gestational sac was “abnormally elongated” and “extends into the cervical canal,” with a short cervix of 1.6 cm (Figure  1A ). She was counseled regarding threatened abortion. Repeat ultrasound at 13 weeks demonstrated similar findings with ongoing viability of a fetus located within the cervix (Figure  1B,C ). Weekly ultrasounds beginning at 16 weeks reported only a stable short cervix (1.2–1.8 cm); she was prescribed vaginal progesterone. At 20 weeks’ gestation, ultrasound demonstrated an anterior low‐lying placenta and concern for PAS. First trimester ultrasound imaging. (A) Elongated 9‐week gestational sac with viable fetus in the cervical canal (star) and empty uterus (arrow). The result was reported initially as a threatened abortion. (B) 13‐week viable fetus and sac within the cervix. (C) 13‐week short cervix with placenta implanted into the dilated cervix (arrow). At our tertiary academic medical center, ultrasound demonstrated normal fetal growth, anatomy, and fluid. Cervical length was 1.2 cm. The amniotic sac and placenta extended into the endocervical canal, suspicious for CEP with abnormal placentation (Figure  2A,B ). Magnetic resonance imaging (MRI) of the abdomen/pelvis confirmed anterior myometrial thinning with concern for placental invasion into the right parametrium (Figure  3 ). Second trimester ultrasound imaging. (A) Transvaginal cervical length performed at 21 weeks’ gestation demonstrates cervical funneling with a residual closed cervical length of 1.2 cm. There is also placenta and amniotic sac extension into the cervical canal. (B) A transabdominal view of the cervix shows features of placenta accreta spectrum (arrow) including myometrial thinning and bridging vessels. Predelivery MRI at 21 weeks’ gestation shows the fetal head and placenta within the cervical canal as well as anterior myometrial thinning without disruption of the bladder wall (arrow). These features are consistent with placenta accreta spectrum. MRI, magnetic resonance imaging. She was counseled regarding termination of pregnancy versus expectant management with a goal for live birth. She opted for termination of pregnancy as she strongly desired future fertility. Consent was obtained for intra‐amniotic digoxin, UAE, and laparotomy with classical hysterotomy to deliver the demised fetus, followed by evaluation of the placenta for removal versus leaving in situ. Possible hysterectomy was also discussed. Fetal demise was induced at 22 weeks via intra‐amniotic digoxin. Three days later, rupture of membranes occurred with uterine cramping the evening prior to planned hysterotomy. She underwent UAE and laparotomy. Intraoperative findings included a bulging hypervascular lower uterus with minimal cervix. The demised fetus was delivered via classical hysterotomy. Portions of placenta were removed with ring forceps but firmly adherent placenta in the dilated cervical canal was left in situ. Given the absence of clinical placenta percreta but ongoing cervical bleeding, a tamponade balloon was placed through the hysterotomy into the cervix, with the catheter draining vaginally. Hemostasis was achieved and she received pitocin and methergine. Her uterus and abdomen were closed. Estimated blood loss was 1500 mL. She received 1 unit of packed red blood cells (pRBCs) and 1 unit of fresh frozen plasma. Postoperatively, she was transitioned from cefazolin to piperacillin‐tazobactam for 24 h due to an isolated fever on POD0. On POD1, oral methergine was stopped and her uterine tamponade balloon was removed. MRI on POD4 demonstrated a heterogeneous upper cervix enlarged to 8.5 cm (Figure  4A ). She was discharged home on POD5 after a normal postoperative course. MRI progression postoperatively. (A) MRI on postop Day 4 reveals an enlarged, heterogeneous upper cervix consistent with the known, adherent products of conception (arrow). (B) MRI at 5 months demonstrated normalization of the uterus and cervix with endometrial thickness of 0.65 cm. MRI, magnetic resonance imaging. She was readmitted on POD15 with fever and tachycardia and diagnosed with endometritis. computed tomography (CT) abdomen/pelvis demonstrated the enlarged, heterogeneous cervix with intracervical fluid and gas foci, which was similar to her POD4 imaging and attributed to retained products. She had a benign abdominal exam, normal lactate, and white blood cell count of 16 × 10 3 /uL. She improved clinically on 48 h of piperacillin‐tazobactam and was transitioned to a 14‐day outpatient course of amoxicillin‐clavulanate. Pelvic exam was deferred to avoid disruption of the retained cervical products. CT abdomen/pelvis at 1 month demonstrated a decrease in the heterogeneous enhancing cervix and her care was transitioned from maternal fetal medicine to gynecology for long‐term care. Beta‐hCG normalized at 6 weeks postpartum and Depo‐Provera was initiated. At 3 months, transabdominal pelvic ultrasound showed near resolution of cervical heterogeneity, with slight irregularity of the lower uterus (Figure  5 ). Depo‐Provera was discontinued per patient preference. MRI at 5 months demonstrated normalization of the uterus and cervix (Figure  4B ). During this surveillance period for placental resorption, pelvic exams were deferred in the absence of concerning symptoms. Three‐month postoperative pelvic ultrasound demonstrates near resolution of the previous cervical heterogeneity. There remained only slight irregularity of the lower uterine segment and upper cervix (arrows). One year postoperatively, she was considering another pregnancy. Preconception transvaginal ultrasound showed small echogenic areas in the lower uterus without increased vascularity (Figure  6 ). She spontaneously conceived after 8 months’ attempt, with normal imaging at 6 weeks’ gestation. Transvaginal cervical lengths were normal (> 3 cm) from 16 to 24 weeks (Figure  7 ). She had an anterior low‐lying placenta that resolved by 27 weeks. There was normal fetal growth, anatomy, and fluid. She delivered a healthy male infant weighing 2815 g via scheduled repeat low transverse Cesarean section at 37 weeks, complicated by uterine atony requiring methergine, tranexamic acid, and a uterine tamponade balloon. Total blood loss was 2200 mL, and she received 2 units of pRBCs. The tamponade balloon was removed on POD1 and she was discharged on POD2. Pelvic ultrasound 1 year after her surgery. (A) Normal uterus and cervix. (B) Doppler without increased vascularity. Transvaginal cervical length measurement at 20 weeks in her G2 pregnancy demonstrates a normal cervix and cervical length of 40.3 mm. Fifteen months later, she reported cyclic pain and a bulge in her midline incision with menses. MRI demonstrated concern for multifocal endometriosis lesions across the rectovaginal space, right round ligament, left ovary, and ventral incision. These findings may represent deeply infiltrating fibrosis from endometrial or residual placental implants. She declined surgical excision and medical suppression. Figure  8 summarizes the case timeline of events. Patient consent for publication was obtained. Timeline of key events for this patient spanning her G1 and G2 pregnancies. CEP, cervical ectopic pregnancy; CT, computed tomography; EBL, estimated blood loss; ED, Emergency Department; MFM, maternal fetal medicine; MRI, magnetic resonance imaging; PAS, placenta accreta spectrum; POD, postoperative day; UAE, uterine artery embolization.

The authors declare no conflicts of interest.