Embarazo triple espontáneo en una paciente con útero didelfo: reporte de un caso y revisión de la bibliografía / Spontaneous triplet pregnancy in a patient with uterus didelphys: report of a case and review of the literature

Resumen ANTECEDENTES: Las anomalías müllerianas implican la desviación de la anatomía normal del aparato reproductor femenino debido a un desarrollo embriológico alterado. Padecer alguna de estas anomalías, en cualquier escenario de su amplio espectro, suele asociarse con infertilidad o desenlaces obstétricos adversos, como la pérdida gestacional temprana o el parto pretérmino. CASO CLÍNICO: Paciente de 21 años, sin antecedentes patológicos relevantes, con dos embarazos finalizados: el primero por cesárea y el segundo por parto (la cesárea se indicó por la presentación podálica, a las 32 semanas). Acudió a un centro de referencia de la ciudad de Medellín, Colombia (Clínica Universitaria Bolivariana) a la primera consulta prenatal a las 17 semanas y 4 días de embarazo, motivada por un cuadro clínico de cólico biliar, sin requerimiento de intervenciones. Se trataba de embarazo triple, monocorial, triamniótico, con diagnóstico de útero didelfo a las 17 semanas y 1 día, con ambos cuellos uterinos reportados en 38 mm. La gestación finalizó a las 28 semanas y 4 días con el nacimiento de tres niños, quienes luego de superar los retos de la prematurez, en la actualidad llevan una vida normal. CONCLUSIONES: Las anomalías müllerianas, en su amplio espectro de manifestación clínica, son una condición que se asocia con una mayor frecuencia de desenlaces adversos materno-perinatales que se incrementan cuando la anomalía se relaciona con un embarazo múltiple. En la bibliografía actual no abundan los reportes de casos que combinen ambas condiciones, ni con recomendaciones oficiales estandarizadas para la atención médica de las pacientes, sobre todo para el escenario de embarazo triple y útero didelfo.

Abstract BACKGROUND: Müllerian anomalies involve deviation from the normal anatomy of the female reproductive tract due to altered embryological development. Having any of these anomalies, in any of their broad spectrum, is often associated with infertility or adverse obstetric outcomes, such as early gestational loss or preterm delivery. CLINICAL CASE: 21-year-old female patient, with no relevant pathological history, with two pregnancies terminated: the first by cesarean section and the second by delivery (the cesarean section was indicated due to breech presentation, at 32 weeks). She attended a referral center in the city of Medellín, Colombia (Clínica Universitaria Bolivariana) for the first prenatal consultation at 17 weeks and 4 days of pregnancy, motivated by a clinical picture of biliary colic, without requiring interventions. The pregnancy was triple, monochorionic, triamniotic, with a diagnosis of didelphic uterus at 17 weeks and 1 day, with both cervix reported at 38 mm. The gestation was terminated at 28 weeks and 4 days with the birth of three infants, who after overcoming the challenges of prematurity, are now living normal lives. CONCLUSIONS: Mullerian anomalies, in their broad spectrum of clinical presentation, are a condition that is associated with an increased frequency of adverse maternal-perinatal outcomes that are increased when the anomaly is associated with multiple pregnancy. The current literature does not abound with case reports combining both conditions, nor with standardized official recommendations for patient care, especially in the setting of triple pregnancy and didelphic uterus.

Septate uterus with double cervix and longitudinal vaginal septum – A report of two cases of rare uterine anomaly

Background: Embryological maldevelopment of the Mullerian or paramesonephric duct results in congenital uterine anomalies. A rare developmental anomaly is a septate uterus with cervical duplication and a longitudinal vaginal septum. This anomaly cannot be explained by the classical unidirectional Mullerian developmental theory and does not fit into American Society for Reproductive Medicine (ASRM) classification of uterine anomalies. The bidirectional Mullerian developmental theory holds good for this type of anomalies and the new ESHRE/ESGE classification for female urogenital tract anomaly (CONUTA) is more suitable for effective categorization, diagnosis, and treatment.Case Report: Two cases of the complete uterine septum with double cervix and longitudinal vaginal septum who presented with dysmenorrhea, dyspareunia, and infertility were diagnosed and managed by hysteroscopy and laparoscopy. The vaginal and the uterine septum were resected. Both the patients had an uneventful post-operative recovery period and were followed up for a period of 1 year. The symptoms such as dysmenorrhea and dyspareunia improved significantly without any effect on the fertility outcome.Conclusion:Very few of such cases are reported in the literature. This rare anomaly is explained by the bidirectional Mullerian developmental theory and the application of the new ESHRE/ESGE classification of the urogenital tract (CONUTA) is more suitable for such anomalies for effective categorization, diagnosis, and treatment.

Operative hysteroscopy-assisted pregnancy termination after failed surgical abortion in missed abortion of woman with complete septate uterus

Structural uterine anomalies in recurrent pregnancy loss

Background: To determine frequency of different structural uterine anomalies in patients with recurrent pregnancy loss.Methods: This observational study was conducted over a period of one and half year at a tertiary care Hospital, included 40 women with recurrent pregnancy loss who underwent combined laparoscopy and hysteroscopy.Results: Twenty-eight patients (70%) had 3 episodes of miscarriage, eight patients (20%) had experienced 4 abortions and three patients (7.5%) had five abortions. Only one patient (2.5%) had six abortions. 32.5% patients had normal hysteroscopy while as 65% patients had no abnormal finding on laparoscopy. Hysteroscopy was abnormal in 67.5% patients with uterine septum (25%) being the most common finding followed by submucous myoma(20%), polyp (12.5%), cervical incompetence (7.5%) and intra uterine adhesions (2.5%). Laparoscopy was abnormal in 35% patients with endometriosis(17.5%) being the most common finding followed by intra pelvic adhesions (15%) and bicornuate uterus (2.5%).Conclusions: Women with recurrent pregnancy loss have increased association with structural uterine anomalies than general population. Both congenital and acquired uterine anomalies are associated with recurrent abortions.

Correlation factor analysis on uterine anomalies in patients with abortion / 中国综合临床

Objective To explore the relationship between the times of abortion and congenital and acquired uterine anomalies.Methods One hundred and eighty-five patients underwent diagnostic hysteroscopy following abortion were selected and diagnosed by hysteroscopy,then divided into congenital and acquired uterine abnormalities according to the morphology and the formation of uterine cavity lesions.The relationship of 1 times,2 times,equal or more than 3 times of different number of abortion and uterine congenital and acquired abnormalities were compared respectively.Results Diagnostic hysteroscopy revealed anomalies uterine cavity was 79.9％ (148/185),of them congenital uterine anomalies was 52.4％ (97/185),and acquired uterine pathologies was the most of 21.1％ (39/185),acquired uterine abnormalities was 21.1％ (39/185),and intrauterine adhesion was the most common of 11.9％ (22/185).Incidence of intrauterine adhesions of natural abortion with 1 times,2 times and 3 times or more were 7.4％ (5/68),14.1％ (11/78) and 15.4％ (6/39).Hysteroscopy showed that the detection rates of normal uterine cavity,congenital abnormalities,acquired uterine and congenital and acquired pathologies in 3 groups were 27.9％ (19/68),51.5％ (35/68),17.6％ (12/68)and 2.9％(2/68);17.9％(14/78),51.3％(40/78),24.3％(19/78) and 6.4％(5/78);10.3％(4/39),56.4％ (22/39),20.5％ (8/39) and 12.8％ (5/39),respectively,the differences were not significant (x2 =8.823,P=0.184).Conclusion Hysteroscopy is a simple and efficient tool in the early diagnosis of congenital and acquired uterine anomalies.Diagnostic hysteroscopy can be performed after the first miscarriage in order to early diagnose and treat congenital and acquired uterine pathologies.

A case of Herlyn-Werner-Wunderlich syndrome: a rare, congenital genitourinary anomaly in a 12-year-old girl

Herlyn-Werner-Wunderlich (HWW) syndrome is a rare, congenital genitourinary anomaly involving the Müllerian and Wolffian structures, and is characterized by the triad of uterine didelphys, obstructed hemivagina, and ipsilateral renal agenesis. It usually presents in adolescent girls in whom hematometrocolpos produces a pronounced mass effect and pain on the side of the obstructed hemivagina. Accurate diagnosis and surgical treatment can be delayed for several months or even years. Here, we report a case of a 12-year-old girl who presented to the emergency department with lower abdominal pain and mass that had lasted for 2 weeks. After the confirmation of HWW syndrome with magnetic resonance imaging, hysteroscopic septostomy was carried out as a definitive treatment. When we evaluate adolescent girls with lower abdominal pain and mass, we should consider the possibility of HWW syndrome.

Clinical characteristics of women with Mullerian anomaly: Twenty years of experience at Asan Medical Center / 대한산부인과학회지

OBJECTIVE: To investigate the clinical characteristics and reproductive outcomes of women with Mullerian anomalies. METHODS: One hundred and eighty-six patients were diagnosed with Mullerian anomalies at the Asan Medical Center from 1990 to 2009 and their clinical characteristics and reproductive outcomes were analyzed. Mullerian anomalies were categorized according to the classification by the American Fertility Society (1988). RESULTS: Mullerian anomaly was noticed in 1 in 1,326 patients (0.075%). Most cases were found in adulthood (84.9%) whereas only 15.1% cases in adolescent or pediatric period. More than 40% of cases were asymptomatic and found incidentally but others suffered from amenorrhea (12.4%), dysmenorrhea (10.8%), abnormal menstruation (10.2%), etc. Most common type of uterine anomalies was uterine didelphys (30.6%), followed by bicornuate uterus (19.4%), Mayer-Rokitansky-Kuster-Hauser syndrome (10.8%), septate uterus (9.1%) and unicornuate uterus (8.6%). On the reproductive outcomes of 251 pregnancies identified, spontaneous miscarriages and preterm labor were quite common (55.8%), and the overall live birth rate was 48.6%. When each anomaly was individually analyzed, the live birth rate was 60.0% in the arcuate uterus and 58.0% in the uterine didelphys. The unicornuate and bicornuate uterus presented a similar chance of having a living child (42.1%, 46.7%), while the septate uterus showed a relatively lower live birth rate (32.4%). CONCLUSION: Although most cases of Mullerian anomalies are diagnosed in adulthood, many patients may suffer from menstrual abnormalities, dysmenorrhea or recurrent miscarriages since adolescence. The reproductive outcomes of the arcuate uterus and uterine didelphys were better, while those of septate uterus were poor in our study.