A very rare case of an ectopic cervical intramural pregnancy

A very rare case of an ectopic cervical intramural pregnancy

CASE REPORT A very rare case of an ectopic cervical intramural pregnancy Teruo Ohtsuka, M.D., Toru Hirata, M.D., Takashi Tsukiyama, M.D., Hiroaki Taka...

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CASE REPORT A very rare case of an ectopic cervical intramural pregnancy Teruo Ohtsuka, M.D., Toru Hirata, M.D., Takashi Tsukiyama, M.D., Hiroaki Takana, M.D., Hideki Kawaguchi, M.D., and Kiminari Terao, M.D. Department of Obsterics, Gynecology, and Perinatology, Miyazaki Prefectural Nobeoka Hospital, Nobeoka City, Miyazaki Prefecture, Japan

Objective: To report a case of a very rare ectopic cervical intramural pregnancy. Design: Case report. Setting: Prefectural hospital. Patient(s): A 22-year-old woman, gravida 1, para 0, was referred to our hospital with the suspicion of a cervical ectopic pregnancy (EP). Pelvic examination revealed an enlarged uterine cervix with no genital bleeding. We found a clear gestational sac (GS) and fetal heart beat in the anterior muscular layer of the uterine cervix by ultrasonography, and confirmed these findings by magnetic resonance imaging (MRI). Intervention(s): We injected methotrexate (MTX) into the GS cavity and around the GS. One week later, the GS was removed surgically without massive bleeding. Main Outcome Measure(s): On the 11th postoperative day, she recovered and was discharged from our hospital. Her menstruation restarted on the 35th postoperative day. Result(s): We have shown a case of a very rare ectopic cervical intramural pregnancy with successful treatment. Conclusion(s): We have explained a case and successful treatment of a very rare ectopic cervical intramural pregnancy with clear GS and fetal heart beat. Our strategy was injecting MTX into the GS cavity and around the GS, then performing an operation to remove the GS. (Fertil Steril 2011;95:291.e11–e13. 2011 by American Society for Reproductive Medicine.) Key Words: Cervical intramural pregnancy, a rare ectopic pregnancy, successful treatment

Cervical pregnancy is a rare form of an ectopic pregnancy (EP). It usually refers to a pregnancy in the uterine cervical canal. We report on the experience of a case of an extremely rare ectopic cervical intramural pregnancy. We found only two case reports (1, 2) of this type of EP in the medical literature. In our patient, the site of pregnancy was located in the middle part of the anterior cervical muscular layer with clear gestational sac (GS) and fetal heart beat (FHB). We successfully treated her with an inject of methotrexate (MTX) into the GS cavity, then performed surgical treatment.

CASE REPORT A 22-year-old woman, gravida 1, para 0, induced abortion 1, was referred to our hospital with suspicion of a cervical pregnancy at 6 weeks and 5 days from her last menstrual period. Pelvic examination revealed a softened and enlarged cervix, which was about the size of a goose egg. Transvaginal ultrasound showed a clear Received January 11, 2010; revised May 3, 2010; accepted May 11, 2010; published online June 18, 2010. T.O. has nothing to disclose. T.H has nothing to disclose. T.T. has nothing to disclose. H.T. has nothing to disclose. H.K. has nothing to disclose. K.T. has nothing to disclose. Reprint requests: Teruo Ohtsuka, M.D., Department of Obsterics, Gynecology, and Perinatology, Miyazaki prefectural Nobeoka Hospital, 2-Chome, 1-Banch, 10 Sinkouji, Nobeoka, Japan (FAX: 011-81-982-26-1930; E-mail: [email protected]).

0015-0282/$36.00 doi:10.1016/j.fertnstert.2010.05.014

GS, 15 mm in diameter, with FHB in the middle portion of the anterior cervical muscular layer. We confirmed this case as a cervical intramural EP by magnetic resonance imaging (MRI) (Fig. 1) and found the GS size to increase by 25-mm increments during several days. Her hCG titer was 28,295 mIU/mL. At 7 weeks and 1 day of gestation, we injected 50 mg of MTX into the GS cavity and in the decidual tissue around the GS under transvaginal ultrasound guidance. Soon after the MTX injection, the FHB stopped. The patient’s hCG titer was 44,833 mIU/mL on the fourth day, and 46,236 mIU/mL on the sixth day after the MTX injection. The ultrasound color Doppler blood flow study showed sufficient blood flow even after the MTX injection. The MRI study revealed that an irregularly shaped GS still in the cervical muscular layer. We decided to perform surgical therapy because she could not be cured by MTX injection alone. On the seventh day after MTX injection, we first tried to remove the EP lesion transvaginally. After incision of the vaginal mucosa at the anterior vaginal fornix, we tried to reveal the cervical muscle layer and the lesion, but the site of the entire lesion was too high to be removed. Therefore, we tried a transabdominal approach instead. After the incision of the vesicouterine peritoneal membrane, we were able to get full exposure to the entire lesion. Then we ligated both uterine arteries to reduce the bleeding during the surgery. The lesion was successfully removed with a small amount of blood loss (300 ml), and the operation time was 113 minutes. Histologic examination showed chorionic tissue

Fertility and Sterility Vol. 95, No. 1, January 2011 Copyright ª2011 American Society for Reproductive Medicine, Published by Elsevier Inc.

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FIGURE 1 A T1-weighted magnetic resonance image (MRI) with gadolinium enhancement (left) shows a clear gestational sac in the anterior muscular layer of the uterine cervix. The T2-weighted MRI (right) shows the cervical canal line and the ectopic pregnancy (EP) lesion in the muscular layer. The gestational sac was 25 mm in diameter, which increased within a few days.

Ohtsuka. Very rare case of ectopic cervical intramural pregnancy. Fertil Steril 2011.

and gestational sacular tissue with a fetus compartment. Her hCG titer decreased to 2,740 mIU/mL on the fourth day, 1,064 mIU/mL on the seventh day, and 323 mIU/mL on the 11th day after operation. On the 11th postoperative day, she was fully recovered and discharged from our hospital. Her menstruation restarted on the 35th postoperative day. Six months after the surgery, we performed a hysteroscopy and a MRI follow-up study. Cervical mucosa was well healed, and we could not find any tract to the IM lesion. The MRI also showed a normal anterior cervical muscle without any lesion.

DISCUSSION We have experienced an extremely rare case of an ectopic cervical intramural pregnancy with clear GS and FHB in the middle portion of the anterior cervical muscular layer. We found only two case reports of cervical intramural EP in medical literature. Ozawa et al. (1) reported the first case, in which the implantation site was in the anterior cervical fibromuscular layer. They found a fine tract between the GS and cervical canal by MRI. After local and systemic chemotherapy with MTX, on the 39th gestational day, necrotic placental tissue was discharged probably through the tract mentioned previously. Taskin et al. (2) reported the second case, in which the implantation site was in the anterior cervical lip. After 1 week from the first visit, the anterior cervical lip was found to be ruptured and chorionic villi was removed easily without active bleeding.

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In our case, the EP site was too deep in the muscular layer to rupture into the vaginal cavity or pass through into the cervical canal. In two previously published reports, the lesions were spontaneously removed through the cervical canal or the vaginal cavity without massive bleeding. In our patient it seemed difficult for the lesion to be removed spontaneously without harmful bleeding. We also thought that it may have taken a long time for the lesion to be absorbed, and the patient would have had to stay in the hospital for a longer period to be prepared for the sudden bleeding that could have led to the need for an emergency operation. This is why, in our case, we decided to perform operative therapy. Antecedent uterine traumatic events involving dilation and curettage, cesarean section, cervical laceration, and instrumentation in the uterine cavity, such as hysteroscopy and IVF and ET, are speculated to be associated with intramural EPs. In our patient, dilation and curettage was the only known predisposing factor. When treating the patient we considered excluding the pregnancy element, to minimize the risk of hemorrhage and to spare fertility. Basically, because there is no path for abortion in cervical intramural EP, we believe that surgical therapy is necessary. After MTX injection and uterine artery ligation, we successfully removed the pregnancy element surgically with a small amount of blood loss. The patient could recover and be discharged quickly. Uterine artery embolization may play a role in the treatment of this type of EP, although at present, uterine artery embolization seems to be used when active bleeding occurs.

Very rare case of ectopic cervical intramural pregnancy

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We have reported a case of an extraordinary ectopic cervical intramural pregnancy. There is no standard in treating this type of

EP because of its rarity. We hope that our case and our strategy becomes a reference when a similar case occurs.

REFERENCES 1. Ozawa N, Takamatsu K, Fujii E, Saito H. Pregnancy implanted in the fibromuscular layer of the cervix. J Reprod Med 2006;51:325–8.

Fertility and Sterility

2. Taskin S, Taskin EA, Cengiz B. Cervical intramural ectopic pregnancy. Fertil Steril 2009;92:395.e5–e7.

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