Mucinous Cystadenoma Arising in a Uterine Isthmocele: A Case Report / Cistadenoma mucinoso surgindo em istmocele uterina: relato de caso

Abstract Isthmocele is a discontinuation of the myometrium at the uterine scar site in a patient with a previous cesarian section (CS). The cause of isthmocele appears to be multifactorial. Poor surgical technique, low incision location, uterine retroflection, obesity, smoking, inadequate healing of scars, and maternal age are possible related factors. Most patients with this condition are asymptomatic. However, women can present with postmenstrual bleeding, pelvic pain, subfertility, dysmenorrhea, infertility, and scar abscess. Brazil has one of the world s highest cesarean section rates. One of the consequences of the rising rate of CS is the isthmocele, an emerging female health problem. Here we report a case of mucinous cystadenoma arising in a uterine isthmocele, a complication, as far as we could investigate, not yet described in the literature.

Resumo Istmocele é a descontinuidade do miométrio no local da cicatriz uterina em paciente com cesariana anterior. A causa da istmocele parece ser multifatorial. Má técnica cirúrgica, baixa localização da incisão, retroflexão uterina, obesidade, tabagismo, cicatrização inadequada de cicatrizes e idade materna são possíveis fatores relacionados. A maioria dos pacientes com esta condição é assintomática. No entanto, as mulheres podem apresentar sangramento pós-menstrual, dor pélvica, subfertilidade, dismenorreia, infertilidade e abscesso cicatricial. O Brasil tem uma das maiores taxas de cesariana do mundo. Uma das consequências da taxa crescente de cesarianas é a istmocele, um problema emergente de saúde feminina. Aqui relatamos um caso de cistoadenoma mucinoso originado em uma istmocele uterina, uma complicação ainda não descrita, até onde pudemos investigar.

Opciones e indicaciones en el manejo del istmocele / Options and indications in the management of isthmocele

INTRODUCCIÓN: El istmocele es un defecto en la cicatrización del sitio de una histerotomía, que puede cursar con sangrado posmenstrual, dolor pélvico, dismenorrea, dispareunia e infertilidad secundaria. Esta patología ha ido incrementando su prevalencia dado el aumento de la tasa de cesáreas en todo el mundo. OBJETIVO: Se realizó una revisión sobre el istmocele y su manejo, presentando sus indicaciones específicas y las complicaciones asociadas a esta patología. MÉTODO: Se llevó a cabo una búsqueda en PubMed, Embase, Scopus y Google Scholar, en la que se encontraron 868 artículos, de los cuales se revisaron 30 al aplicar los criterios de inclusión y exclusión. DISCUSIÓN: El istmocele es cada vez más frecuente. Tiene una prevalencia cercana al 60% posterior a la realización de una cesárea y aumenta hasta valores del 100% con tres de ellas. Los métodos diagnósticos más utilizados son la ecografía transvaginal y la histerosonografía. Su abordaje es habitualmente quirúrgico, aunque existe la posibilidad de intentar tratamiento médico en algunos casos. CONCLUSIONES: Es necesario determinar el grosor miometrial para poder establecer un plan de manejo adecuado. Además, se ameritan estudios que realicen un seguimiento a largo plazo y que aporten mayor evidencia para la realización de cada procedimiento. Después de clasificar el tipo de defecto, el tratamiento quirúrgico del istmocele se debe ofrecer a pacientes sintomáticas y a aquellas con defectos grandes y que desean mantener la fertilidad.

INTRODUCTION: The isthmocele is a defect in the healing of the site of a hysterotomy, which can present with post-menstrual bleeding, pelvic pain, dysmenorrhea, dyspareunia and secondary infertility. This pathology has been increasing its prevalence given the increase in the rate of cesarean sections worldwide. OBJECTIVE: A review will be carried out of the isthmocele and its management, presenting its specific indications and the complications associated with this pathology. METHOD: A search was carried out in databases such as PubMed, Embase, Scopus and Google Scholar, finding a total of 868 articles, of which 30 of them were reviewed when applying the inclusion and exclusion criteria. DISCUSSION: Isthmocele is an increasingly frequent pathology, having a prevalence of 60% after performing a cesarean section and increasing to 100% with 3 of them. There are multiple diagnostic methods, mainly transvaginal ultrasound and sono-hysterosonography. The approach to this pathology is usually surgical, although there is the possibility of trying medical treatment in some cases. CONCLUSIONS: It is necessary to determine the myometrial thickness in order to establish an adequate management plan. Additionally, long-term follow-up studies are warranted and provide more evidence for the performance of each procedure. After classifying the type of defect, surgical treatment of the isthmocele should be offered to symptomatic patients or those with large defects and who desire future fertility.

Associação entre a técnica de fechamento uterino na cesárea e a istmocele: uma visão global / Association between uterine closure technique after cesarean and isthmocele: an overview

A istmocele (ou "defeito cicatricial de cesariana") é uma alteração anatômica na parede uterina responsável por formar um "nicho", que é visualizado como uma área hipoecoica por histerografia ou por outros métodos de imagem, devido a uma cicatrização inapropriada de uma cesárea anterior. Essa modificação leva a complicações ginecológicas e obstétricas. Este estudo pretendeu analisar, entre os fatores de risco, a relação da istmocele com a técnica de fechamento uterino, comparando a sutura em camada simples com a dupla. Foi realizada uma revisão de literatura por meio de pesquisa bibliográfica em diferentes bases de dados eletrônicos. Foram encontrados 31 artigos, todavia apenas 13 estudos foram incluídos no presente estudo após avaliação criteriosa. A técnica de fechamento uterino é um dos fatores de risco possível de prevenir, sendo que a sutura em camada simples é associada a maior chance de se desenvolver a istmocele. A sutura de camada simples (ancorada) incluindo o endométrio está relacionada a menor espessura miometrial residual, associada a probabilidade de ruptura uterina e a istmocele, que a camada dupla não ancorada com a exclusão do endométrio.(AU)

The isthmocele is an anatomical alteration in the uterine wall responsible for forming a "niche", which is visualized as a hypoechoic area by hysterography or other imaging methods, due to inappropriate healing of a previous cesarean section. This modification leads to gynecological and obstetric complications. This study aimed to analyze among the risk factors the relation of isthmocele and the uterine closure technique comparing the single- and double-layer suture. It was conducted an electronic based search in different electronic databases. The research led to the retrieval of 31 articles; however only 13 studies were included in the present research after careful reading. The uterine closure technique is one of the possible risk factors to be modified; the single-layer suture is associated with a greater chance of developing isthmocele. The locked single-layer suture including the decidua is related to the decreased residual myometrial thickness, associated with the likelihood of uterine rupture and the isthmocele, than the double-layer unlocked excluding the decidua.(AU)

Isthmocele: from risk factors to management / Istmocele: de fatores de risco ao manejo

Abstract Objective The aim of the present study was to perform a comprehensive review of the literature to provide a complete and clear picture of isthmocele-a hypoechoic area within themyometriumat the site of the uterine scar of a previous cesarean section-by exploring in depth every aspect of this condition. Methods A comprehensive review of the literature was performed to identify the most relevant studies about this topic. Results Every aspect of isthmocele has been studied and described: pathophysiology, clinical symptoms, classification, and diagnosis. Its treatment, both medical and surgical, has also been reported according to the actual literature data. Conclusion Cesarean section is the most common surgical procedure performed worldwide, and one of the consequences of this technique is isthmocele. A single and systematic classification of isthmocele is needed to improve its diagnosis and management. Further studies should be performed to better understand its pathogenesis.

Resumo Objetivo O objetivo do presente estudo foi realizar uma revisão abrangente da literatura a fim de fornecer um quadro completo e claro da istmocele-uma área hipoecoica dentro domiométrio no local da cicatriz uterina de uma cesariana anterior- aprofundando todos os aspectos desta condição Métodos Uma revisão abrangente da literatura foi realizada para identificar os estudos mais relevantes sobre este tema. Resultados Todos os aspectos da istmocele foram estudados e descritos: fisiopatologia, sintomas clínicos, classificação e diagnóstico. Os tratamentos médico e cirúrgico também foram relatados de acordo com os dados reais da literatura. Conclusão A cesárea é o procedimento cirúrgico mais comum realizado em todo o mundo, e uma das consequências desta técnica é a istmocele. Uma classificação única e sistemática da istmocele é necessária para melhorar seu diagnóstico e manejo. Novos estudos devem ser realizados para melhor entender sua patogênese.

Istmocele: una revisión sistemática de la literatura / Istmocele: a systematic review of literature

Resumen OBJETIVO: Revisar la bibliografía de la prevalencia, factores de riesgo, síntomas, diagnósticos y tratamiento de las pacientes con istmocele. MÉTODO: Búsqueda electrónica en las bases de datos: PubMed, EMBASE y Google Scholar. Se utilizaron los siguientes términos, palabras y sus combinaciones: "Cesarean section defect, uterine niche, isthmocele, uterine sacculation, uterine diverticulum, uterine pouch, isthmocele diagnosis, segmentocele y isthmocele treatment". La variable primaria estudiada fueron los síntomas asociados con el istmocele. Las variables secundarias: prevalencia, factores de riesgo, diagnóstico y tratamiento. RESULTADOS: Se reunieron 549 artículos de los que se eliminaron 288 por duplicidad y 228 no cumplieron los criterios de inclusión; al final solo se analizaron 33 artículos. El istmocele tiene una prevalencia de 15 a 84% en mujeres con antecedente de cesárea. Su incidencia se correlaciona directamente con la cantidad de cesáreas previas. Su aparición puede ser sintomática o asintomática. La manifestación clínica más común es el sangrado uterino anormal, que sucede en 28.9 a 82% de los casos. Incluso 88% se diagnostican en el ultrasonido transvaginal. La histeroscopia quirúrgica se asoció con disminución de los síntomas en 56.9 a 100%. CONCLUSIONES: El istmocele suele identificarse de manera fortuita en el ultrasonido transvaginal y casi siempre es asintomático. Puede ocasionar sangrado uterino anormal e infertilidad secundaria. Su prevalencia depende del método diagnóstico utilizado. La histeroscopia es el método de tratamiento más efectivo.

Abstract OBJECTIVE: Review the literature on the prevalence, risk factors, symptoms, diagnoses and treatment of patients with isthmocele. METHOD: An electronic search was performed using the following databases: PubMed, EMBASE and Google Scholar. The following terms, words and their combinations were used: "Cesarean section defect, uterine niche, isthmocele, uterine sacculation, uterine diverticulum, uterine pouch, isthmocele diagnosis, segmentocele y isthmocele treatment". The primary outcome was the symptoms associated with a cesarean scar defect. The secondary outcomes were prevalence, risk factors, diagnosis and treatment of istomocele. RESULTS: 549 articles were collected, of which 288 were eliminated due to duplication and 228 did not meet the inclusion criteria; In the end, only 33 articles were analyzed. A prevalence of 15 to 84% was found in women with a previous caesarean section. The prevalence of this alteration is correlated with the number of cesarean sections; the greater the number of caesarean sections, the greater the risk of developing an isthmocele. Its presence can be symptomatic or asymptomatic. The most common symptom is abnormal uterine bleeding, occurring in a 28.9% to 82% of the patients. Up to 88% of cases are diagnosed by a transvaginal ultrasound. A surgical hysteroscopy was associated with a 56.9% to a 100% improvement of symptoms. CONCLUSIONS: Isthmocele is commonly identified incidentally through a transvaginal ultrasound and is usually asymptomatic. It can cause abnormal uterine bleeding and secondary infertility. Its prevalence depends on the diagnostic method used. A surgical hysteroscopy is the most effective treatment method.

Importancia ginecológica del istmocele ¿cómo y cuándo tratar? / Gynecological importance of the isthmocele ¿When and how to treat?

Resumen OBJETIVO: determinar la incidencia de defectos en el cierre de la histerorrafia, sus síntomas y analizar las conductas terapéuticas. MATERIALES Y MÉTODOS: estudio observacional, prospectivo y comparativo de pacientes con defectos en el cierre de la histerorrafia, atendidas en la Clínica de Colposcopia del Centro Estatal de Oncología del Estado de Sonora entre el 1 de enero de 2013 y el 31 de diciembre de 2015. Criterios de inclusión: pacientes con citología anormal y antecedente de cesárea, a quienes se practicaron: colposcopia, endocervicocolposcopia e histeroscopia. Criterios de exclusión: problemas técnicos que impidieron la observación del canal endocervical o la histeroscopia, pacientes posmenopáusicas, y las que tuvieran diagnóstico colposcópico de cáncer invasor. RESULTADOS: se estudiaron 120 pacientes con antecedente de cesárea, que se practicó en diferentes centros hospitalarios, de éstas se excluyeron 25 y quedaron 95 casos: 62 (65%) del grupo A (con defectos en el cierre de la histerorrafia) y 33 (35%) del grupo B (sin defectos en el cierre de la histerorrafia). De los 62 casos con defectos en el cierre de la histerorrafia, 30 (50%) fueron en el cierre de la histerorrafia tipo cúpula, 20 (31%) tipo divertículo y 12 (19%) dehiscencia total. Hubo manchado menstrual en 73% de las pacientes, polimenorrea en 31% y dismenorrea en 33%. Solo 20% de las pacientes sintomáticas refirieron los síntomas como relevantes. El aumento de la vascularidad y el sangrado se encontraron en las pacientes con más síntomas.

Abstract OBJECTIVE: The Cesarean Scar Defect (CSD) has been underestimated in frequency and importance. Is necessary to know its incidence, the symptoms, and analyze new therapeutic procedures. MATERIAL AND METHODS: Observational, prospective and comparative study of patients with defects in the closure of hysterorrhaphy, attended at the Colposcopy Clinic of the State Oncology Center of the State of Sonora between January 1, 2013 and December 31, 2015. Inclusion criteria: Patients with abnormal cytology and previous cesarean section, who were: colposcopy, endocervicocolposcopy and hysteroscopy. Exclusion criteria: technical problems that prevented observation of the endocervical canal or hysteroscopy, postmenopausal patients, and those with a colposcopic diagnosis of invasive cancer. RESULTS: We found in 65.2%, spotting in a 73.3%, polymenorrhea at 31.3%, and to dysmenorrhea in 32.3%. Only 20% of the symptomatic patients referred the symptom as Relevant. We found "CSDs producers of bleeding" that are related to the patients more systematic. CONCLUSIONS: The cesarean scar defects are frequent cause of gynecological symptomatology that is irrelevant most of the time. A small number of patients needs treatment, we found CSD that produce bleeding, this cases present relevant symptomatology and require treatment. It is necessary to take this into account to choose the best therapeutic approach.

Prevalencia de istmocele en el Hospital de San José de Bogotá, Colombia / Prevalence of isthmocele at the San José Hospital in Bogotá, Colombia

Objetivo: Describir la prevalencia de istmocele como hallazgo incidental en pacientes con antecedente de cesárea y síntomas clínicos asociados. Métodos: Estudio descriptivo de corte transversal, mediante muestreo no probabilístico por conveniencia, en pacientes con antecedente de cesárea, programadas para histeroscopia entre noviembre de 2014 y marzo de 2015, en el servicio de cirugía endoscópica ginecológica del Hospital San José de Bogotá, Colombia. Resultados: 42 pacientes fueron elegibles para el estudio por su antecedente de cesárea y todas fueron incluidas. La frecuencia de istmocele fue 83,3% en la histeroscopia, con similar localización en istmo y cérvix. La principal característica clínica presentada por las pacientes fue hemorragia uterina anormal (85,7%), mientras que la menos frecuente fue infertilidad (7,1%). En las pacientes con presencia de istmocele se observó una mayor prevalencia de dismenorrea (65,7% vs. 42,9%), antecedente de 2 o más cesáreas (60% vs. 42,9%) y cesárea de urgencia (54,3% vs. 28,6%) comparadas con el grupo de pacientes sin istmocele, en este último grupo se advirtió que el 100% de las pacientes no tenían antecedente de trabajo de parto previo. En mujeres con antecedente de cesárea y presencia de síntomas como sangrado uterino anormal, dismenorrea, dolor pélvico, infertilidad y dispareunia, la frecuencia de istmocele diagnosticado por histeroscopia fue mayor del 80%. Conclusión: El istmocele se debe a la cicatrización anómala uterina posterior a una cesárea, se requieren otros estudios para determinar no solo la prevalencia sino los factores protectores que reduzcan su incidencia para tener un impacto positivo en este tipo de pacientes.

Objective: To describe the prevalence of isthmocele as an incidental finding in patients with a history of cesarean section and associated clinical symptoms. Methods: Descriptive cross-sectional study using nonprobabilistic sampling for convenience in patients with a history of cesarean section, scheduled for hysteroscopy between November 2014 and March 2015, in the gynecological endoscopic surgery service of the Hospital San José de Bogotá, Colombia. Results: 42 patients were eligible for the study because of their previous cesarean section and all were included. The frequency of isthmocele was 83.3% in hysteroscopy, with similar localization in the isthmus and cervix. The main clinical characteristic presented by the patients was abnormal uterine bleeding (85.7%), while the less common was infertility (7.1%). A higher prevalence of dysmenorrhea (65.7% vs. 42.9%) was observed in patients with isthmocele, a history of 2 or more cesareans (60% vs. 42.9%) and an emergency cesarean section (54, 3% vs. 28.6%) compared to the group of patients without isthmocele, in the latter group it was noted that 100% of the patients had no previous history of labor. In women with a history of cesarean section and presence of symptoms such as abnormal uterine bleeding, dysmenorrhea, pelvic pain, infertility and dyspareunia, the frequency of isthmocele diagnosed by hysteroscopy was greater than 80%. Conclusion: Isthmocele is due to abnormal uterine cicatrization after cesarean section, other studies are required to determine not only the prevalence but also the protective factors that reduce its incidence to have a positive impact on this kind of patients.