Ectopic pregnancy is a common cause of morbidity and mortality in women of reproductive age especially in resource deprived areas worldwide. Cervical ectopic pregnancy is a rare, life threatening form of ectopic pregnancy which needs a high index of suspicion for diagnosis, thus adding a complex twist to the dilemma faced by the obstetrician in resource deprived areas. A case of a cervical ectopic pregnancy which presented in a resource deprived area in a region in Ghana is discussed, and the difficulties encountered in diagnosis and management of this lifethreatening condition are outlined.Keywords: Ectopic pregnancy, cervical, resource deprived areas, difficult diagnosis, managemen

Mouhajer, Mohammed

Obed, Samuel

Okpala, Amalachukwu M.

English

AJOL - African Journals Online

Case Report                                                                                                                                                                                                                                                                                                                                                                   www.ghanamedj.org  Volume 50 Number 2 June 2016 94 Cervical Ectopic Pregnancy in Resource Deprived Areas: A Rare and Diffi-cult Diagnosis  Mohammed Mouhajer1, Samuel Obed2 and Amalachukwu M. Okpala3  Ghana Med J 2017; 51(2): 94-97 http://dx.doi.org/10.4314/gmj.v51i2.8  1Cape Coast Teaching Hospital, Central Region, Ghana 2Department of Obstetrics and Gynaecology, Korle-Bu Teaching Hospital, Accra, Ghana. 3Department of Breast Surgery, Abertawe Bro Morgannwg University Health Board, Wales, UK  Corresponding author: Dr Mohammed Mouhajer    Email: hamude@hotmail.com Conflict of interest: None declared  SUMMARY Ectopic pregnancy is a common cause of morbidity and mortality in women of reproductive age especially in re-source deprived areas worldwide. Cervical ectopic pregnancy is a rare, life threatening form of ectopic pregnancy which needs a high index of suspicion for diagnosis, thus adding a complex twist to the dilemma faced by the obste-trician in resource deprived areas. A case of a cervical ectopic pregnancy which presented in a resource deprived area in a region in Ghana is discussed, and the difficulties encountered in diagnosis and management of this life threatening condition are outlined.  Keywords: Ectopic pregnancy, cervical, resource deprived areas, difficult diagnosis, management  INTRODUCTION Ectopic pregnancy is a common cause of morbidity and sometimes mortality in women of reproductive age. The incidence of ectopic pregnancy ranges from 1 in 30 de-liveries in Ghana, which falls within the range of 1 in 44 and 1 in 21 deliveries seen in developing countries, as compared to 1 in 280 to 1 in 233 for developed coun-tries.1 Management of ectopic pregnancy depends on site of presentation, and whether ruptured or unruptured. Cervical ectopic pregnancy is a rare, life threatening form of ectopic pregnancy with an incidence of 1 in 9,000 deliveries.2 It adds a complex twist to the dilem-ma faced by the obstetrician in resource deprived areas in the diagnosis of an ectopic pregnancy.  We present a case of cervical ectopic pregnancy pre-senting in a 35 year old multiparous patient at a district hospital in Ghana.  CASE REPORT A 35 year old grand multipara (Para 8) presented with bleeding per vaginam for 3 days and symptoms of anaemia. The last menstrual period was about 2 months prior to presentation. There was no associated ab-dominal pain.    The past medical/surgical history was unremarkable, though patient had complained of heavy periods for three months prior to presentation. Previous deliveries had been per vaginam.  On examination, vital signs were stable, though she was markedly pale. Abdominal exam was unremarkable. Pelvic exam with a speculum revealed friable nodular masses on the anterior lip of the cervix which bled easi-ly on touch. Abdominopelvic scan showed no intrauter-ine/ extra uterine pregnancies. Blood tests revealed a haemoglobin of 5.9g/dl.  Urine B-HCG was positive. On further discussion and review, a tentative diagnosis of cervical tumour was made.   The patient was transfused and the bleeding subsided subsequently. Patient was discharged to be readmitted in the next 4 days for a biopsy. Patient however defaulted, and was readmitted 6 weeks later with heavy bleeding per vaginam and severe anaemia. Patient was rescusci-tated with multiple blood transfusions, and reassessed again. Repeat transabdominal scan showed a bulky uterus with absent intrauterine gestational sac and nor-mal adnexa.  A vaginal examination revealed an en-larged mass on the anterior lip of the cervix with a spurting vessel.   Case Report                                                                                                                                                                                                                                                                                                                                                                   www.ghanamedj.org  Volume 50 Number 2 June 2016 95 A diagnosis of cervical ectopic gestation was made. An emergency total abdominal hysterectomy was done. Findings were a 16 week sized empty uterus, and what appears to be a cervical ectopic gestation containing products of conception which were intimately attached to the cervix and could not be peeled off (Figure 1).   Post-operatively, patient recovered fully and was dis-charged on the 8th post-operative day. She was however lost to follow up again and thus no histopathology re-port was obtained.    Figure 1 Cervical ectopic pregnancy       DISCUSSION Cervical pregnancy is a rare form of ectopic pregnancy in which the blastocyst implants in the lining of the en-docervical canal. It accounts for less than 1% of ectopic pregnancies with an incidence of 1:9,000 deliveries.2 In our facility, there has been no prior record of a cervical ectopic gestation. Risk factors include previous dilata-tion and curettage, previous caesarean section delivery, rapid transit of fertilized ovum due to unreceptive en-dometrium, pre pregnancy smoking, pelvic inflammato-ry disease and in vitro fertilization.2,3 Asherman’s syn-drome, prior instrumentation or therapeutic abortion use, infertility, and prior ectopic pregnancies have also been implicated.3 Majority of women with cervical pregnancy have a low parity.3 In this case, the patient was para 8 and all were delivered per vaginam. There was no previous history of dilatation and curettage, nor use of any contraceptive method.  Cervical ectopic pregnancy has been classified accord-ing to site of origin: isthmico-cervical, pure cervica, cervico-isthmic and cervico-isthmic-corporal pregnan-cy.4 Clinical presentation is usually painless profuse first trimester vaginal bleeding.  Lower abdominal cramping is conspicuously absent and is found in less than one third of patients 3. Pain without bleeding is rare.2 Cervical pregnancy rarely progresses beyond the twentieth week, however, exceptions do occur when it has progressed to a live birth.5,6  Physical examination usually reveals a soft cervix that is disproportionately enlarged in comparison to the uterus, a partially open external os, and haemorrhage on ma-nipulation of the cervix.7 These signs are not diagnostic, and if suspicion of cervical pregnancy arises radiologi-cal evaluation is mandatory.7  Cervical ectopic pregnancy was first described in 1817, and was named as such in 1860.2 In 1911 Rubin came up with a histopathological criteria for diagnosis of cer-vical pregnancy.8 1. Cervical glands must be opposite the placental attachment. 2. Placental attachment to the cervix must be situ-ated below the entrance of the uterine vessels or below the peritoneal reflection of the anteri-or and posterior surfaces of the uterus. 3. Fetal elements must be absent from the corpus uteri. 4. The attachment of the placenta to the cervix must be intimate.  Rubin’s criteria required pathologic examination of the excised uterus. However, if the uterus is preserved, the-se criteria cannot be applied. Thus, Duckman suggested the following diagnostic criteria9: 1. A dilated, thin-walled cervix containing histo-logic evidence of gestation. 2. A patulous external os. 3. Small and firm corpus uteri with normal size internal os resting ontop of dilated cervix.  The most widely accepted clinical criteria are those de-fined by Paalman and McElin.10 These are: 1. Amenorrhea followed by uterine bleeding without cramping pain. 2. A softened and disproportionately enlarged cervix equal to or larger than the corporal por-tion of the uterus (an hour glass-shaped uterus). 3. Products of conception entirely confined with-in, and firmly attached to the endocervix. 4. A snug internal os. 5. Partially open external os.  As mentioned above, ultrasonographic investigations are mandatory for confirmation of suspected cervical ectopic pregnancy.   Int. Os Cervix + POC Bisected Uterus Case Report                                                                                                                                                                                                                                                                                                                                                                   www.ghanamedj.org  Volume 50 Number 2 June 2016 96 Ultrasonographic criteria with transvaginal ultrasound are stated below as follows7: 1. Diffuse amorphous intrauterine echoes. 2. Empty uterus (absence of an intrauterine preg-nancy) 3. Uterine enlargement. 4. Characteristic enlargement of the cervix con-taining products of conception.  The main differential diagnosis of a cervical pregnancy is a cervical abortion.10 Use of colour Doppler sonogra-phy to detect peritrophoblastic blood flow to distinguish cervical ectopic pregnancy from an aborting intrauterine pregnancy is important. Vaginal sonography can also help differentiate the two due to the sliding motion of the latter against the endocervical canal as compared to the former which is implanted into the cervix.7 Distin-guishing features between the two conditions have been suggested by Duckman in 1957.11  Management can be conservative or surgical. The major goals are to minimize hemorrhage and preserve future fertility when necessary.12 However, this depends on the state of the patient on presentation to the facility.  Conservative management includes the use of intramus-cular methotrexate. This is effective in 80 – 90% of cas-es of early cervical pregnancy.10  Criteria for the use of methotrexate include a hemodynamically stable patient as well as ectopic size of 3cm or less.10,13  Surgical management involves dilatation and evacua-tion, hysteroscopy and hysterectomy as a last resort. The main complication of dilatation and evacuation is a high incidence of severe hemorrhage which can be reduced by preoperative measures like transvaginal ligation of cervical branches of the uterine arteries, cervical encer-clage, angiographic uterine artery embolization.10   Hysteroscopic resection is a potentially safe and effec-tive option as it allows direct visualization for resection of the ectopic gestation, and simultaneous ablation of bleeding vessels.13   The two surgical options above are used when fertility sparing is being considered and in cases where there is no life threatening hemorrhage. In cases of massive hemorrhage in a hemodynamically unstable patient, emergency hysterectomy is the best option.  Difficulties in diagnosis and management of cervical ectopic pregnancy in the West African setting Patients with cervical ectopic pregnancy are at high risk of severe, potentially life-threatening hemorrhage if diagnosis is delayed.  Conservative management is effective and safe, but its success depends on the diagnostic accuracy of the initial ultrasound.2  In our case, there was some initial confusion about the diagnosis, as the assessment was carried out by junior doctors. The rarity of the condition does not help mat-ters, thus it is seldom suspected or diagnosed before surgery as one needs a very high level of suspicion.11 The initial ultrasound was also inaccurate. It was done trans-abdominally by a radiographer, and generally, ultrasound results tend to be operator – specific, more so when not done by a radiologist. Also, as the diagnosis of a cervical ectopic pregnancy was not thought of, efforts were not made to pick up more details to allow inclu-sion or otherwise from the above mentioned diagnostic criteria. On hind sight, we believe the clinical and radio-logical criteria would have been met.  These are the conditions that are prevalent in district hospitals in developing countries where the clinics are manned by non-specialists, in addition to the paucity of diagnostic equipment. Even when the diagnostic equip-ment are available, there is also a lack of skilled person-nel with the necessary experience to operate them. Thus when such rare conditions present, it is difficult to make a diagnosis, leading to emergency radical operations as occurred in our case.  Management of cervical ectopic gestation is also a prob-lem in resource deprived areas. Health provision is still essentially a cash and carry system, as the national health insurance scheme does not cover all the costs of treatment and patients can hardly afford to buy medica-tion. These patients commonly present with hemor-rhage, and the acquisition of blood and blood products is a problem for patients due to the shortage in blood banks. The patients also have to pay for processing of the blood product before it is issued out to them.   In Ghana, the national demand for blood is more than 250,000 annually, but the national collection rate is far less than 50%.14 Conservative management is usually out of the question due to the emergency presentation as happened in this case. Methotrexate is quite expensive as well, and patients may not be able to afford it since it is not under the National Health Insurance Scheme list of drugs.  Dilatation and evacuation is also a tricky option for re-source deprived areas. There is a lack of equipment for embolisation of the cervical/ uterine arteries preopera-tively, and hysteroscopic equipment in the rural areas.   Case Report                                                                                                                                                                                                                                                                                                                                                                   www.ghanamedj.org  Volume 50 Number 2 June 2016 97 Although there are various methods for arresting hemor-rhage such as cervical cerclage and tamponade of the cervix before the consideration of ligation of arter-ies/hysterectomy, one also considers the amount of blood loss involved as well as the availability of blood products for transfusion.15  All this unfortunately minimizes the possibility of fertil-ity sparing in women of low parity who present with cervical ectopic pregnancies.  In our case, this patient received a total of 7 units of blood, and had an emergency total abdominal hysterec-tomy due to the ongoing torrential blood loss. Fertility sparing was not an issue in this case due to the fact that the patient was multiparous, and consented to the pro-cedure.  Finally, histopathological confirmation of the diagnosis is very difficult, as there are usually no pathologists in district hospitals. Patients usually have to carry the samples themselves and travel to the capital city, Accra, for the services of a pathologist, of which patients can-not afford. Also, when it comes to obtaining results, patients usually have to wait for well over a month, and some patients lose interest and never return for them. In our case, we lost the patient to follow up, so we cannot even be sure the sample was sent for histopathology. Therefore, apart from our clinical findings, we only have a photograph to show as evidence.  CONCLUSION Cervical ectopic pregnancy is a rare condition whose diagnosis requires a high level of suspicion, and a high level of experience, both from the obstetrician and the radiologist. Early diagnosis is essential for successful conservative management thus maintaining fertility, avoiding blood transfusion and radical surgical interven-tions.   Diagnosis and conservative management of this condi-tion in a resource deprived area is difficult due to lack of experienced personnel and supportive equipment in rural areas. This leads ultimately to radical interventions which have a higher rate of morbidity.  REFERENCES 1. Obed, SA. Diagnosis of Unruptured Ectopic Preg-nancy is still uncommon in Ghana. Ghana Med J 2006; 40(1): 3-7. 2. Kameswari, S. A case of cervical ectopic pregnan-cy: successful therapy with methotrexate. J Obstet Gynaecol India 2012; 62(S1): S1 –S3. 3. Heer JS, Chao DK, McPheeters RA. Cervical Ec-topic Pregnancy. West J Emerg Med 2012; 13 (1): 125 -126. 4. David MP, Bergman A, Delighdish L. Cervic-isthmic pregnancy carried to term. Obstet Gynecol 1980; 56; 247. 5. Pisarski TS. Cervical pregnancy. Br J Obstet Gy-naecol 1960; 67:759. 6. Mitrani, A. Cervical pregnancy ending in a live birth. Br J Obstet Gynaecol 1973; 80:761. 7. Nasrolahi SH, Pilevari SH, Neghab N. Cervical ectopic pregnancy: successful treatment with meth-otrexate. Pak J Med Sci 2008; 24 (6): 883 – 886. 8. Rubin IC. Cervical pregnancy. Surg Gynecol Obstet 1911; 13:625. 9. Duckman, S. Cervical pregnancy. Am J Obstet Gy-necol 1951; 62: 1381. 10. Varghese U, Fajardo A, Gomathinayagam T. Cer-vical pregnancy. Oman Med J 2008; 23:53 – 54. 11. Nickens OJ. Cervical Pregnancy: A case report. J Natl Med Assoc 1962; 54 (1): 91 – 93. 12. Kofinas JD et al. Hysteroscopic Removal of Cervi-cal Ectopic Pregnancy Following Failed Intramus-cular/Intra-Sac Methotrexate: A Case Report. J Gy-necol Surg 2012; 28 (5): 369 -371. 13. Slaughter JL, Grimes DA. Methotrexate therapy – Non surgical management of ectopic pregnancy. West J Med 1995; 162: 225 – 228. 14. Kweku. “Ghana needs over 250,000 units of blood annually” Ghana Health Nest 30 Sept 2013. Web. 19 Sept 2014. http://gocreatestudio.com/healthnest/2013/09/30/ghana-needs-250000-units-blood-annually/ 15. Sharma A et al. Cervical intramural pregnancy: Report of a rare case. Niger Med J 2013 Jul – Aug; 54(4): 271 – 273.✪ 

Cervical Ectopic Pregnancy in Resource Deprived Areas: A Rare and Difficult Diagnosis

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Cervical Ectopic Pregnancy in Resource Deprived Areas: A Rare and Difficult Diagnosis

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