**2. Historical review of cervical ectopic pregnancy**

According to Thomsen and Johansen, the first case of cervical pregnancy was reported in 1817 by Sir Everard Home, who found an early ovum in the cervical canal during postmortem examination. Thereafter, more and more cases were reported soon after Karl Freiherr von Rokitansky described two cases in 1860 in the German literature. The maternal mortality rate early in the 1900s was exceptionally high mainly due to hemorrhage and sepsis (66% according to Hofsatter; 43% according to Zangemeister and Schilling; 13% according to Concetti), and was estimated to be an average of 30%[9].

However, this rate is not considered to have actually been this high, because of the limited ability of exact diagnosis in those days. That is, patients underwent evacuation of the uterus based on incorrect diagnosis, consequently suffering from massive hemorrhage, which was treated by hysterectomy. In 1946, Schneider defined "distal ectopic pregnancy" as a pregnancy in which the fetus resides in the cervical canal. He classified it into three categories by implantation site: (1) 'pure' cervical ectopic pregnancy; (2) isthmico-cervical pregnancy; and (3) endometrio-isthmico-cervical pregnancy[10]. With the probable

Fertility-Preserving Surgery for Cervical Ectopic Pregnancy, from Past to Present 227

In addition to these two cases, few cases have addressed efforts to determine a successful conservative therapy—one case by Whittle in 1976[8], four cases by Materacaru in 1968[19],

The summary of the procedures for these cases are as follows. 1) The approach is vaginal. 2) The urinary bladder is moved upwards through a transverse incision of the anterior vaginal wall to make visible the whole anterior cervix. 3) Bleeding is controlled by clamping each side of the cervix to occlude the lateral cervical blood vessel. 4) The anterior cervical wall is incised longitudinally upwards from the external os to the internal os. 5) If the conceptive products have been implanted in the anterior wall, the local cervical wall is excised with the ectopic fetus, and both sides are sutured; if the implantation site is posterior, complete curettage is performed under direct visualization, usually requiring several sutures for the torn sites of the thin wall. 6) The procedure is achieved by removing the hemostasis clamp and packing the vaginal and cervical canal with gauze. During the procedure, blood

transfusion of >1000 ml is usually necessary to maintain the patient's circulation.

salpingo-oophorectomy because of a recurrent tubo-ovarian abscess.

blood loss was 1200 ml, necessitating whole-blood transfusion.

Although such procedures involve a vaginal approach, abdominal surgery was also attempted during the same period. In 1969, Nelson applied ligation of the bilateral internal iliac arteries in two cases to perform successful curettage[20], following the idea of

The first case was a 17-year-old woman (gravida 1, para 0) who consulted a doctor because of low abdominal pain and abnormal vaginal bleeding. Although a 12-week pregnancysized pelvic mass was confirmed, the pregnancy test was negative. Therefore, exploratory laparotomy was indicated and it showed normal sized uterine corpus that had elevated out of the pelvis by a 10-cm soft cystic enlargement of the cervix. Cervical pregnancy was diagnosed. A large amount of necrotic placental tissue and an old clot was removed by vaginal curettage with 250 ml of blood loss after bilateral internal iliac artery ligation under laparotomy. Three years later, she became pregnant which ended in incomplete infected abortion. The following year, she received total abdominal hysterectomy and bilateral

The second case was a 33-year-old woman (gravida 3, para 2) who consulted a doctor with profuse vaginal bleeding at week 11 of gestation. With a diagnosis of threatened abortion, curettage was attempted. The first introduction of forceps into the cervical canal caused sudden loss of over 300 ml of blood, and further evacuation brought on uncontrollable hemorrhage. Laparotomy confirmed cervical pregnancy and bilateral internal iliac artery ligation was applied, followed by evacuation of the placental tissue by curettage. Estimated

Shinagawa stated that satisfactory hemostasis could not be achieved by internal iliac artery ligation, based on his reports of 19 cases which all received a hysterectomy after attempts to save the uterus; he did not indicate the number of cases that underwent this procedure. As an alternative method to bilateral internal iliac artery ligation, Akashi et al. applied bilateral uterine artery ligation by the vaginal approach to stop bleeding in a case of massive hemorrhage. Akashi et al. further reviewed 29 successful cases of uterine conservation in Japan until 1976. These cases included curettage only in eight patients, curetting plus removal of the gestational sac in one, conservative treatment only in one (the method was not specified), curettage plus internal artery ligation in one, curettage plus cervical cerclage in one, cervicotomy only in one (the approach was not specified), abdominal cervicotomy in

and one case by Farghaly et al. in 1980[6].

Dodeck[21].

inclusion of distal ectopic pregnancy other than true cervical pregnancy or other unconfirmed cases, Baptisti estimated the mortality rate to be only 6% of cases published between 1945 and 1953 and stated that this remarkable decline in the mortality rate was due to the development of blood transfusion in modern obstetrics[11].

Shinagawa, in his 10 years of experience, reported in 1969 about 19 cervical pregnancy cases, all of which resulted in abdominal hysterectomy after attempted vaginal treatment to save the uterus. He expressed his surprise for the discrepancy in the frequency of cases between the United States and Japan[12]. In the United States, a little over 80 cases had been reported up until 1967[13]. Sheldon et al. experienced two cases at the Mayo Clinic over a 15-year period, an incidence of approximately 1 in 16,000 pregnancies[7], while Paalman and McElin found only five cases in a series of 47,974 pregnancies at two American hospitals over a 10 year period—an incidence of 0.01%[14]. On the other hand, in Japan, in addition to Shinagawa's 19 cases of approximately 19,000 pregnancies reported at his university hospital and affiliated hospitals between 1958 and 1967, at least 119 cases had been reported throughout Japan between 1953 and 1967[12], suggesting the estimated incidence of the cervical ectopic pregnancy in Japan to be 0.1% (1:1000 pregnancies). Considering that no nulliparous woman was in his series and in 13 of the 19 cases of antecedent pregnancy were interrupted artificially, Shinagawa supposed that the difference in incidence was due to the higher number of legal abortions at that time in Japan. Therefore, the true incidence of cervical ectopic pregnancy is unknown, and comparison between countries is difficult because of the differences in cultures and eras.

Summarizing the past treatments in the literature, Thomsen and Johansen stated that most of the cases were treated based on incorrect original diagnosis[15]. Attempts to evacuate the uterus digitally or instrumentally usually produced violent hemorrhage, and in many cases it was so severe as to necessitate hysterectomy. Several authors reportedly controlled the bleeding by packing, sometimes with fibrin foam, or by amputating the cervix. Although utero-tonic agents are generally used against bleeding after delivery, Danforth pointed out that such agents are ineffective against hemorrhage as the open vessels could not be closed by contraction of the thin-walled, distended cervix, which contains few contractile elements[16]. Schneider and Drezin[17] and Steinbiss[18] also noted that tamponade may provide initial success of hemostasis, but a severe secondary hemorrhage may nevertheless occur up to six weeks later necessitating hysterectomy. By the 1950s, clinicians attempted to conserve the uterus during the treatment of cervical ectopic pregnancy with the establishment of blood transfusion methods[11].
