Annals of African Medicine Vol. 12, No. 4; 2013
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Case Report
Website: www.annalsafrmed.org DOI: 10.4103/1596-3519.122696 PMID: ******
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Successful pregnancy in one horn of a bicornuate uterus Adewale Samson Adeyemi, Oluseyi Olaboyede Adeyi Atanda, Adebayo Duyile Adekunle Department of Obstetrics and Gynecology, Ladoke Akintola University of Technology, Ogbomoso, Oyo State, Nigeria
Correspondence to: Dr. Adewale Samson Adeyemi, Department of Obstetrics and Gynaecology, Ladoke Akintola University of Technology Teaching Hospital, P M B - 4008, Ogbomoso, Oyo State , Nigeria. E‑mail: [email protected]
Abstract Normal development of the female reproductive tract involves a series of complex processes characterized by the differentiation, migration, fusion, and subsequent canalization of the Müllerian system. Uterine anomalies result when these processes are interrupted. Bicornuate uterus is a unification defect of the Müllerian ducts, and it is estimated to represent 10-39% of Müllerian duct anomalies. Pregnancies in bicornuate uterus are usually considered high-risk because of association with poor reproductive outcomes, such as pregnancy loss, preterm birth, malpresentations, and fetal deformity. Routine cervical cerclage and Strassman metroplasty have been advocated as treatment for this anomaly. We report a case of successful pregnancy in one of the horns of a bicornuate uterus managed in our unit. Keywords: Adverse pregnancy outcome, successful term pregnancy, uterus bicornuate
Résumé Développement normal de l'appareil reproducteur féminin comporte une série de processus complexes, caractérisés par la différenciation, la migration, fusion et canalisation ultérieur du système de canaux. Les anomalies utérines se produire lorsque ces processus sont interrompus. Utérus bicorne est un défaut d'unification des conduits canaux, et on estime à représenter 10-39 % des anomalies de conduit de canaux. Grossesse dans l'utérus bicorne est habituellement considérés à haut risque en raison de la liaison avec les mauvais résultats de la reproduction, tels que la perte de la grossesse, prématurité, malpresentations et malformation foetale. Le cerclage du col utérin systématique et Strassman metroplasty ont été préconisés dans le traitement de cette anomalie.Nous rapportons le cas d'une grossesse réussie dans l'une des cornes d'un utérus bicorne gérés dans notre unité. Mots-clés: Grossesse à terme avec succès, utérus bicorne
Introduction Congenital uterine anomalies result from the abnormal formation, fusion or resorption of Müllerian ducts during fetal life.[1] Normal development of the female reproductive tract involves a series of complex processes characterized by the differentiation, migration, fusion, and subsequent canalization of the Müllerian system.[2] Uterine anomalies result when these processes are interrupted. Vol. 12, October-December, 2013
The prevalence of uterine anomalies varies, depending on the population and the diagnostic method. A prevalence of 1-10% was found in an unselected population, 2-8% in subfertile women, and 5-30% of women with a history of miscarriage.[3] Bicornuate uterus is a unification defect of the Müllerian ducts, and it is estimated to represent 10-39% of Müllerian duct anomalies.[4] In most cases, a bicornuate uterus is incidentally discovered Annals of African Medicine
Adeyemi, et al.: Uterus bicornuate: Successful term pregnancy
Case Report
gestational age revealed a viable pregnancy in the right horn of the bicornuate uterus, and was compatible with the estimated gestational age. She had two doses of anti‑tetanus toxoid, and also two doses of intermittent preventive treatment with sulphadoxin/pyrimethamine as anti‑malaria prophylaxis. Repeat obstetric ultrasound at 34 weeks gestational age revealed no abnormality.
A 32-year-old female, booked G3P1+1 (not alive) referred from University College Hospital, Ibadan, because she relocated from Ibadan to Ogbomoso, where our institution is located. She was 5 weeks and 4 days amenorrheic at presentation.
The antenatal period was uneventful until 36 weeks gestational age when she started having preterm contractions. She was evaluated and admitted for bed rest. Vaginal examination revealed normal vaginal and a firm, smooth closed cervical os.
She was diagnosed to be having bicornuate unicollis uterus by ultrasonography in the course of evaluation for primary infertility in 2008.
At 37 completed gestational age, she had elective caesarean section under spinal anesthesia. The operative findings were: a clean pelvis, a gravid right horn with poorly formed, lower uterine segment, and smaller left horn of a bicornuate uterus [Figure 2], scanty clear liquor, a live male infant with birth weight 2.8 kg, apgar scores 6 and 9 at 1 and 5 minutes, respectively; the placenta was antero‑fundal, the uterine tubes and the ovaries were grossly normal. The operative blood loss was 500 ml.
when the pelvis is imaged. The most common symptomatic presentation is with early pregnancy loss and cervical incompetence.[4] We present a case of successful pregnancy in one of the horns of a bicornuate uterus managed in our unit.
In 2009, she had intrauterine fetal death at term and ultrasonography revealed the dead fetus to be in the left horn of a bicornuate uterus [Figure 1]. She subsequently had caesarean delivery and was delivered of a stillborn infant at a Christian mission hospital. In 2010, she had spontaneous abortion at estimated gestational age of 12 weeks, and ultrasonography revealed the non‑viable conceptus to be in the left horn of the bicornuate uterus. The abortion was complete, and there was no need for uterine evacuation. At our institution, the index pregnancy was confirmed by serum pregnancy test. She was booked for antenatal care, and the routine investigation at booking revealed a hemoglobin concentration of 9.5 g/dl, Rhesus positive blood group O, AA hemoglobin genotype, hepatitis B surface antigen negative, non‑reacting retroviral screening, and the urinalysis was within the normal limits. Ultrasonography at 12 weeks
Figure 1: Ultrasound scan showing the gestation in the left horn of the bicornuate uterus, and empty right horn (arrowed)
Annals of African Medicine
Pediatrician assessment of the baby revealed that the fore foot of both legs were abducted and supinated, and high arched; there was also overriding of the second and fourth toes bilaterally, and mild pseudo‑tallipes. The baby was admitted into the special care baby unit for observation. The postoperative period was uneventful, and the patient was discharged home on 7th postoperative day after removal of stitches. The postnatal follow‑up revealed no abnormality and the previously noted abnormality in the baby had resolved. The mother was counseled on family
Figure 2: The exteriorized bicornuate uterus showing the Caesarean incision on the right horn of the bicornuate uterus after the delivery of the baby. The right horn that contained the baby is obviously bigger than the left
Vol. 12, October-December, 2013
Page | 253
Adeyemi, et al.: Uterus bicornuate: Successful term pregnancy
planning methods and encouraged to opt for a method as soon as possible, and she was discharged from postnatal clinic.
Discussion
Page | 254
Bicornuate uterus was previously thought to be associated with infertility,[5] but recent studies have not confirmed such an association.[6] However, pregnancies in a bicornuate uterus are usually considered high‑risk that require extra monitoring because of association with poor reproductive potential. Such adverse reproductive outcomes that has been found to be associated with the anomaly are recurrent pregnancy loss, preterm birth, malpresentations, and fetal deformity.[7] The patient presented had a previous first trimester pregnancy loss, and the successful pregnancy being presented was associated with fetal limb deformity which was found to have resolved during postnatal follow‑up. The fetal limb deformity might result from prolonged pressure on the limbs due to lack of space within the uterine horn where the fetal development took place. The inadequate room for rotation can also be the explanation for the fetal malpresentations that has been found to be associated with bicornuate uterus.[8] The cephalic presentation assumed by the fetus at the beginning of the third trimester was maintained till term in the case presented. Preterm delivery due to cervical incompetence had been found to be associated with bicornuate uterus,[7] and this has led some workers to advocate routine cervical cerclage for pregnant bicornuate uterus and other uterine anomaly.[9] The patient being presented had carried two pregnancies to term without cervical cerclage, although the first pregnancy ended in intrauterine fetal death, which was unlikely to be related with the uterine anomaly. Several cases of successful term singleton and twins pregnancies had been reported without cervical cerclage.[3,5,8] Therefore, there may be need for a randomized controlled trial to determine the appropriateness of routine cervical cerclage for pregnant bicornuate uterus. Bicornuate uterus is associated with adverse pregnancy outcome rather than infertility. The patient being discussed was diagnosed to have
Vol. 12, October-December, 2013
bicornuate uterus in the course of evaluation for infertility, therefore, bicornuate uterus may be an incidental finding in subfertile population, and this may account for its small contribution to infertility of all uterine anomalies.[3] As in the case presented, several cases of successful pregnancies in bicornuate uterus had been reported without surgical correction of the anomaly,[5,8] therefore, the Strassman metroplasty should be reserved for cases of bicornuate uterus with recurrent preterm delivery that is not responding to cervical cerclage. Successful term pregnancies had been reported following such surgical intervention.[10]
References 1.
Moore KL, Persaud TV, Torchia MG. The urogenital system. In: Before We Are Born: Essentials of Embryology and Birth Defects. 7th ed. Philadelphia: Saunders/Elsevier; 2008. p. 162‑189. 2. Ammesse LS, Pfaff‑Amesse T. Congenital anomalies of the reproductive tract. In: Falcone T, Hurd WW, editors. Clinical Reproductive Medicine and Surgery. 1 st ed. New York: Mosby; 2007. p. 171‑190. 3. Raga F, Bauset C, Remohi J, Bonille‑Musa F, Simon C, Pellicar A. Reproductive impact of Congenital Müllerian anomalies. Hum Reprod 1997;12:2277‑81. 4. Nahum GG. Uterine anomalies. How common are they, and what is their distribution among subtypes? J Reprod Med 1998;43:877‑87. 5. Shwiqing M, Xuming B, Jingha L. Pregnancy and its outcome in women with malformed uterus. Chin Med Sci J 2002;17:242‑5. 6. Proctor JA, Hang AF. Recurrent first trimester pregnancy loss is associated with uterine septum but not with bicornuate uterus. Fertil Steril 2003;80:1212‑5. 7. Chan YY, Javaprakasan K, Tan A, Thanton JG, Coomarasamy A, Raine‑Fenning NJ. Reproductive outcomes in women with congenital uterine anomalies: A systematic review. Ultrasound Obstet Gynecol 2011;38:371‑82. 8. Ekwempu CC, Ogbe EA. Persistent breech presentation in a bicornuate uterus: A case Report. J Med Trop 2011;13:145‑6. 9. Golan A, Langer R, Wexter S, Segey E, Niv D, David MP. Cervical cerclage‑its role in the pregnant anomalous uterus. Int J Fertil 1990;35:164‑70. 10. Papp Z, Mezei G, Gavai M, Hupuezi P, Urbanesek J. Reproductive performance after transabdominal metroplasty: A review of 157 consecutive cases. J Reprod Med 2006;51:544‑52. Cite this article as: Adeyemi AS, Atanda OA, Adekunle AD. Successful pregnancy in one horn of a bicornuate uterus. Ann Afr Med 2013;12:252-4. Source of Support: Nil, Conflict of Interest: None declared.
Annals of African Medicine
Copyright of Annals of African Medicine is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
Access this article online Quick Response Code:
Case Report
Website: www.annalsafrmed.org DOI: 10.4103/1596-3519.122696 PMID: ******
Page | 252
Successful pregnancy in one horn of a bicornuate uterus Adewale Samson Adeyemi, Oluseyi Olaboyede Adeyi Atanda, Adebayo Duyile Adekunle Department of Obstetrics and Gynecology, Ladoke Akintola University of Technology, Ogbomoso, Oyo State, Nigeria
Correspondence to: Dr. Adewale Samson Adeyemi, Department of Obstetrics and Gynaecology, Ladoke Akintola University of Technology Teaching Hospital, P M B - 4008, Ogbomoso, Oyo State , Nigeria. E‑mail: [email protected]
Abstract Normal development of the female reproductive tract involves a series of complex processes characterized by the differentiation, migration, fusion, and subsequent canalization of the Müllerian system. Uterine anomalies result when these processes are interrupted. Bicornuate uterus is a unification defect of the Müllerian ducts, and it is estimated to represent 10-39% of Müllerian duct anomalies. Pregnancies in bicornuate uterus are usually considered high-risk because of association with poor reproductive outcomes, such as pregnancy loss, preterm birth, malpresentations, and fetal deformity. Routine cervical cerclage and Strassman metroplasty have been advocated as treatment for this anomaly. We report a case of successful pregnancy in one of the horns of a bicornuate uterus managed in our unit. Keywords: Adverse pregnancy outcome, successful term pregnancy, uterus bicornuate
Résumé Développement normal de l'appareil reproducteur féminin comporte une série de processus complexes, caractérisés par la différenciation, la migration, fusion et canalisation ultérieur du système de canaux. Les anomalies utérines se produire lorsque ces processus sont interrompus. Utérus bicorne est un défaut d'unification des conduits canaux, et on estime à représenter 10-39 % des anomalies de conduit de canaux. Grossesse dans l'utérus bicorne est habituellement considérés à haut risque en raison de la liaison avec les mauvais résultats de la reproduction, tels que la perte de la grossesse, prématurité, malpresentations et malformation foetale. Le cerclage du col utérin systématique et Strassman metroplasty ont été préconisés dans le traitement de cette anomalie.Nous rapportons le cas d'une grossesse réussie dans l'une des cornes d'un utérus bicorne gérés dans notre unité. Mots-clés: Grossesse à terme avec succès, utérus bicorne
Introduction Congenital uterine anomalies result from the abnormal formation, fusion or resorption of Müllerian ducts during fetal life.[1] Normal development of the female reproductive tract involves a series of complex processes characterized by the differentiation, migration, fusion, and subsequent canalization of the Müllerian system.[2] Uterine anomalies result when these processes are interrupted. Vol. 12, October-December, 2013
The prevalence of uterine anomalies varies, depending on the population and the diagnostic method. A prevalence of 1-10% was found in an unselected population, 2-8% in subfertile women, and 5-30% of women with a history of miscarriage.[3] Bicornuate uterus is a unification defect of the Müllerian ducts, and it is estimated to represent 10-39% of Müllerian duct anomalies.[4] In most cases, a bicornuate uterus is incidentally discovered Annals of African Medicine
Adeyemi, et al.: Uterus bicornuate: Successful term pregnancy
Case Report
gestational age revealed a viable pregnancy in the right horn of the bicornuate uterus, and was compatible with the estimated gestational age. She had two doses of anti‑tetanus toxoid, and also two doses of intermittent preventive treatment with sulphadoxin/pyrimethamine as anti‑malaria prophylaxis. Repeat obstetric ultrasound at 34 weeks gestational age revealed no abnormality.
A 32-year-old female, booked G3P1+1 (not alive) referred from University College Hospital, Ibadan, because she relocated from Ibadan to Ogbomoso, where our institution is located. She was 5 weeks and 4 days amenorrheic at presentation.
The antenatal period was uneventful until 36 weeks gestational age when she started having preterm contractions. She was evaluated and admitted for bed rest. Vaginal examination revealed normal vaginal and a firm, smooth closed cervical os.
She was diagnosed to be having bicornuate unicollis uterus by ultrasonography in the course of evaluation for primary infertility in 2008.
At 37 completed gestational age, she had elective caesarean section under spinal anesthesia. The operative findings were: a clean pelvis, a gravid right horn with poorly formed, lower uterine segment, and smaller left horn of a bicornuate uterus [Figure 2], scanty clear liquor, a live male infant with birth weight 2.8 kg, apgar scores 6 and 9 at 1 and 5 minutes, respectively; the placenta was antero‑fundal, the uterine tubes and the ovaries were grossly normal. The operative blood loss was 500 ml.
when the pelvis is imaged. The most common symptomatic presentation is with early pregnancy loss and cervical incompetence.[4] We present a case of successful pregnancy in one of the horns of a bicornuate uterus managed in our unit.
In 2009, she had intrauterine fetal death at term and ultrasonography revealed the dead fetus to be in the left horn of a bicornuate uterus [Figure 1]. She subsequently had caesarean delivery and was delivered of a stillborn infant at a Christian mission hospital. In 2010, she had spontaneous abortion at estimated gestational age of 12 weeks, and ultrasonography revealed the non‑viable conceptus to be in the left horn of the bicornuate uterus. The abortion was complete, and there was no need for uterine evacuation. At our institution, the index pregnancy was confirmed by serum pregnancy test. She was booked for antenatal care, and the routine investigation at booking revealed a hemoglobin concentration of 9.5 g/dl, Rhesus positive blood group O, AA hemoglobin genotype, hepatitis B surface antigen negative, non‑reacting retroviral screening, and the urinalysis was within the normal limits. Ultrasonography at 12 weeks
Figure 1: Ultrasound scan showing the gestation in the left horn of the bicornuate uterus, and empty right horn (arrowed)
Annals of African Medicine
Pediatrician assessment of the baby revealed that the fore foot of both legs were abducted and supinated, and high arched; there was also overriding of the second and fourth toes bilaterally, and mild pseudo‑tallipes. The baby was admitted into the special care baby unit for observation. The postoperative period was uneventful, and the patient was discharged home on 7th postoperative day after removal of stitches. The postnatal follow‑up revealed no abnormality and the previously noted abnormality in the baby had resolved. The mother was counseled on family
Figure 2: The exteriorized bicornuate uterus showing the Caesarean incision on the right horn of the bicornuate uterus after the delivery of the baby. The right horn that contained the baby is obviously bigger than the left
Vol. 12, October-December, 2013
Page | 253
Adeyemi, et al.: Uterus bicornuate: Successful term pregnancy
planning methods and encouraged to opt for a method as soon as possible, and she was discharged from postnatal clinic.
Discussion
Page | 254
Bicornuate uterus was previously thought to be associated with infertility,[5] but recent studies have not confirmed such an association.[6] However, pregnancies in a bicornuate uterus are usually considered high‑risk that require extra monitoring because of association with poor reproductive potential. Such adverse reproductive outcomes that has been found to be associated with the anomaly are recurrent pregnancy loss, preterm birth, malpresentations, and fetal deformity.[7] The patient presented had a previous first trimester pregnancy loss, and the successful pregnancy being presented was associated with fetal limb deformity which was found to have resolved during postnatal follow‑up. The fetal limb deformity might result from prolonged pressure on the limbs due to lack of space within the uterine horn where the fetal development took place. The inadequate room for rotation can also be the explanation for the fetal malpresentations that has been found to be associated with bicornuate uterus.[8] The cephalic presentation assumed by the fetus at the beginning of the third trimester was maintained till term in the case presented. Preterm delivery due to cervical incompetence had been found to be associated with bicornuate uterus,[7] and this has led some workers to advocate routine cervical cerclage for pregnant bicornuate uterus and other uterine anomaly.[9] The patient being presented had carried two pregnancies to term without cervical cerclage, although the first pregnancy ended in intrauterine fetal death, which was unlikely to be related with the uterine anomaly. Several cases of successful term singleton and twins pregnancies had been reported without cervical cerclage.[3,5,8] Therefore, there may be need for a randomized controlled trial to determine the appropriateness of routine cervical cerclage for pregnant bicornuate uterus. Bicornuate uterus is associated with adverse pregnancy outcome rather than infertility. The patient being discussed was diagnosed to have
Vol. 12, October-December, 2013
bicornuate uterus in the course of evaluation for infertility, therefore, bicornuate uterus may be an incidental finding in subfertile population, and this may account for its small contribution to infertility of all uterine anomalies.[3] As in the case presented, several cases of successful pregnancies in bicornuate uterus had been reported without surgical correction of the anomaly,[5,8] therefore, the Strassman metroplasty should be reserved for cases of bicornuate uterus with recurrent preterm delivery that is not responding to cervical cerclage. Successful term pregnancies had been reported following such surgical intervention.[10]
References 1.
Moore KL, Persaud TV, Torchia MG. The urogenital system. In: Before We Are Born: Essentials of Embryology and Birth Defects. 7th ed. Philadelphia: Saunders/Elsevier; 2008. p. 162‑189. 2. Ammesse LS, Pfaff‑Amesse T. Congenital anomalies of the reproductive tract. In: Falcone T, Hurd WW, editors. Clinical Reproductive Medicine and Surgery. 1 st ed. New York: Mosby; 2007. p. 171‑190. 3. Raga F, Bauset C, Remohi J, Bonille‑Musa F, Simon C, Pellicar A. Reproductive impact of Congenital Müllerian anomalies. Hum Reprod 1997;12:2277‑81. 4. Nahum GG. Uterine anomalies. How common are they, and what is their distribution among subtypes? J Reprod Med 1998;43:877‑87. 5. Shwiqing M, Xuming B, Jingha L. Pregnancy and its outcome in women with malformed uterus. Chin Med Sci J 2002;17:242‑5. 6. Proctor JA, Hang AF. Recurrent first trimester pregnancy loss is associated with uterine septum but not with bicornuate uterus. Fertil Steril 2003;80:1212‑5. 7. Chan YY, Javaprakasan K, Tan A, Thanton JG, Coomarasamy A, Raine‑Fenning NJ. Reproductive outcomes in women with congenital uterine anomalies: A systematic review. Ultrasound Obstet Gynecol 2011;38:371‑82. 8. Ekwempu CC, Ogbe EA. Persistent breech presentation in a bicornuate uterus: A case Report. J Med Trop 2011;13:145‑6. 9. Golan A, Langer R, Wexter S, Segey E, Niv D, David MP. Cervical cerclage‑its role in the pregnant anomalous uterus. Int J Fertil 1990;35:164‑70. 10. Papp Z, Mezei G, Gavai M, Hupuezi P, Urbanesek J. Reproductive performance after transabdominal metroplasty: A review of 157 consecutive cases. J Reprod Med 2006;51:544‑52. Cite this article as: Adeyemi AS, Atanda OA, Adekunle AD. Successful pregnancy in one horn of a bicornuate uterus. Ann Afr Med 2013;12:252-4. Source of Support: Nil, Conflict of Interest: None declared.
Annals of African Medicine
Copyright of Annals of African Medicine is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
