Rapidly evolving pelvic lymphangioleiomyomatosis (LAM) mimicking bilateral hydrosalpinx: report of a rare case and literature review.

Lymphangioleiomyomatosis (LAM) represents a rare neoplasm affecting almost exclusively women of reproductive age. This condition mainly affects the lungs, but extrapulmonary locations such as the pelvis and the retroperitoneum are possible. Clinical evaluation and ultrasound imaging are usually non-specific, and the diagnosis is obtained through surgical excision and histopathological examination. We report a very rare case of abdominal LAM in a young female patient. A thorough literature review of this rare condition with emphasis on gynecologic implications will be presented. The patient was referred for gynecologic consultation due to pelvic pain and infertility. Unfortunately, despite prompt diagnosis and treatment, the course of the disease was severe and led to patient's exitus in a short time. We encountered an extremely rare deadly pathology mimicking a very common gynecologic condition. The gynecologist must always be alert of possible unexpected conditions that will require prompt attention.

In-office Hysteroscopic Treatment of Herlyn-Werner-Wunderlich Syndrome: A Case Series.

Herlyn-Werner-Wunderlich syndrome (HWWs) is a rare congenital malformation of the female urogenital track characterized by a triad consisting of didelphys uterus, obstructed hemivagina, and ipsilateral renal agenesis. We report 5 consecutive cases of patients diagnosed with HWWs treated in our center. Imaging studies with 2-dimensional/3-dimensional ultrasound and abdominopelvic magnetic resonance imaging were obtained to confirm the diagnosis. The treatment consisted of a 1-step surgical in-office hysteroscopic incision of the vaginal septum using 5 Fr hysteroscopic bipolar electrodes. At the follow-up visit, between 1 and 2 months after the initial procedure, the patients underwent a diagnostic vaginoscopy with excision of exceeding vaginal tissue if needed, performed with a vaginal hysteroscopic approach. In-office hysteroscopic treatment of patients diagnosed with HWWs is a safe and effective, minimally invasive treatment modality that provides symptomatic relief and preserves fertility, avoiding the cost and risks of the use of general anesthesia in an operating room setting. We recommend shifting the management of this challenging condition to the office setting.

Long-Term Reproductive Outcomes after Hysteroscopic Treatment of Dysmorphic Uteri in Women with Reproductive Failure: An European Multicenter Study.

OBJECTIVE: To evaluate the long-term reproductive outcomes in patients with dysmorphic uterus treated by hysteroscopic metroplasty with miniaturized instruments. DESIGN: Retrospective multicenter cohort study. SETTING: Tertiary care university hospitals. PATIENTS: The study was conducted on 214 women with a dysmorphic uterus (T-shaped, infantilis, or other type of dysmorphic uterus according to the European Society of Human Reproduction and Embryology and the European Society for Gynaecological Endoscopy classification system) with history of primary unexplained infertility (group 1) or repeated (>2) early miscarriages (group 2). Dysmorphic uteri were diagnosed by office hysteroscopy and 3-dimensional transvaginal ultrasound (3D-TVS). INTERVENTIONS: All patients underwent in office hysteroscopic metroplasty using a continuous-flow hysteroscope with a 5 Fr operating channel introduced into the uterine cavity using the vaginoscopic approach. Longitudinal incisions were performed on the fibromuscular constriction rings in the isthmic area and in some cases on the other uterine walls with a 5 Fr bipolar electrode or scissors. At the end of the procedure, an antiadhesive gel was applied into the uterine cavity to minimize adhesion formation. Postsurgical assessment of the uterine cavity was carried out through office hysteroscopy and 3D-TVS. All patients were followed for at least 24 months. MEASUREMENTS AND MAIN RESULTS: The metroplasty was completed in all cases, resulting in a significant increase of uterine cavity volume (100%) and optimization of uterine morphology in 211 of 214 women (98.6%). After 60 months, the overall clinical pregnancy rate was 72.9% (n = 156/214), and the live birth rate was 80.1% (n = 125/156). Specifically, 74 of 156 women (47.4%) conceived spontaneously (with a median time to pregnancy of 5.5 months), of whom 32.4% had previously failed 1 or more attempts at in vitro fertilization/intracytoplasmic sperm injection. CONCLUSION: Our long-term follow-up data demonstrate that the hysteroscopic correction of dysmorphic uteri may result in a high live birth rate in women suffering from unexplained infertility or repeated miscarriages.

Resectoscopic Metroplasty with Uterine Septum Excision: A Histologic Analysis of the Uterine Septum.

STUDY OBJECTIVE: To evaluate the histology of the uterine septum after its complete hysteroscopic excision. DESIGN: Case series. SETTING: Second Gynecological and Obstetric Unit and Pathological Anatomy Department of the University of Bari, Italy. PATIENTS: Thirty-five patients aged between 25 and 41 years who were diagnosed with uterine septum by 3-dimensional ultrasound per the European Society for Human Reproduction and Embryology/European Society of Gastrointestinal Endoscopy 2013/Salim 2003 criteria. In addition, office hysteroscopy was performed to define the anatomy of the uterine cavity and to exclude the presence of other endometrial pathologic conditions. INTERVENTIONS: Operative hysteroscopic septum resection was performed. The septum was initially incised with an "L-shape" bipolar electrode with a 5-mm bipolar mini-resectoscope (KARL STORZ SE & Co. KG, Tuttlingen, Germany). Then, using the bipolar loop, 2 triangles of the septum were excised in parallel, obtaining uninterrupted entire septum-long strips from the fundus to the apex of the septum. These strips were immediately removed from the uterus and reassembled in vitro to reconstruct a macroscopic, 3-dimensional structure of the septum for complete morphologic and histologic evaluations. MEASUREMENTS AND MAIN RESULTS: Patients presented with an average body mass index of 24.8 kg/m2and were all nulliparous. Histologic evaluation of the uterine septa showed a different conformation of the muscle bundles along the septum. Muscle cells in the apex and edges of the septum were arranged in nodules circumscribed by a thin area of collagen fibers. Medium-sized vessels were distributed in the collagen fibers around the muscle cells. Only few capillary vessels were present in the muscle nodules. This pattern was very similar to the cell arrangement in leiomyomas. In the core of the septa, near the base, the muscle bundles showed a linear course with concurrent collagen fibers and vessels. All the aforementioned characteristics were consistently present in every patient. On high-power histologic fields (200×), the muscle portion accounted for 48.3% ± 1.8% (mean, 6%) area in the apex and borders to 48.5% ± 1.3% (mean, 6%) area in the core. Collagen fibers accounted for 27.1% ± 1.1% (mean, 4%) area in the apex and borders to 26.7% ± 1.3% (mean 5%) area in the core. CONCLUSION: By removing the septum as a whole structure, this study allowed us to redefine the concept of the septum as a complex structure according to the islands of muscle fibers irregularly arranged in vertex, in a context of collagen tissue and similar to the structure of myomas. It appears to deeply involve the anterior and posterior uterine walls, resembling a "reverse letter H."

Comparison of endometrial polyp recurrence in fertile women after office hysteroscopic endometrial polypectomy using two widely spread techniques.

AIM: To compare the recurrence of benign endometrial polyps after office hysteroscopic polypectomy performed with a bipolar electrode (BE) or a small diameter hysteroscopic tissue removal system (HTRs). METHODS: From July 2018 to December 2019 we evaluated the charts of 114 asymptomatic fertile women who underwent office hysteroscopic polypectomy, 1 year before, for a single large benign endometrial polyp (size between 10 and 20 mm) using a 4 mm continuous flow hysteroscope with a BE or a 5 mm HTRs. Patients, divided into two groups according to surgical procedure, each performed exclusively by one expert gynecologist, were scheduled for a 12-month postoperative transvaginal sonography to evaluate the recurrence of endometrial polyps. RESULTS: Forty-eight women of the BE group and 42 of the HTRs group were considered for the 1-year transvaginal sonography follow-up. Five polyps were identified in the BE group and three in the HTRs group (5/48 vs 3/42, P = n.s.). All polyps were removed hysteroscopically (in three out of five and in two out of three cases, respectively, in the same places of the previous polypectomy) and evaluated as 'benign' by the pathologist. CONCLUSION: Office hysteroscopic endometrial polypectomy with small HTRs compared to BE revealed at a 1-year follow-up no difference in terms of complete removal and recurrence of polyps. HTRs polypectomy resulted in less pain and significantly quicker time of procedure compared to BE. This data should be kept in mind for patient comfort any time hysteroscopic polypectomy is planned in an office setting.

Age at menarche and clinical outcomes following medically assisted reproduction (MAR): a cohort study.

Medically assisted reproduction (MAR) technologies have advanced rapidly, but in contrast to the specificity of modern approaches, they provide limited effectiveness in the management of the infertile couple. The purpose of this study was to assess the possible relationship between age at menarche and MAR outcomes of clinical pregnancy, live birth and the adverse incident of miscarriage, and to determine the offspring sex ratio according to age at menarche. In a cohort of 254 infertile couples who underwent 426 IVF/ICSI cycles, statistical analysis was performed by applying Student's t-test, chi-square test, and logistic regression models, adequately in the respective parameters and outcomes. The results indicated a strong association of age at menarche with the outcomes of clinical pregnancy (p = .0007) and live birth (p < .0001), especially by applying a threshold of 12 years in the first occurrence of menstruation (p = .0019 for clinical pregnancy, p < .0001 for live birth), also demonstrating a negative effect for earlier menarche that acts in parallel with the increasing age of the woman. Calculation of sex ratio demonstrated a tendency towards female offspring close to the age at menarche of 12 years. Age at menarche could serve as a surrogate parameter for reproductive potential towards personalized management of infertility.

Chronic Endometritis, a Common Disease Hidden Behind Endometrial Polyps in Premenopausal Women: First Evidence From a Case-Control Study.

STUDY OBJECTIVE: To investigate the correlation between endometrial polyps (EPs) and chronic endometritis (CE). DESIGN: Single-center retrospective case-control study. SETTING: Academic center. PATIENTS: A total of 480 premenopausal women with abnormal uterine bleeding (AUB) were enrolled. Group A included 240 women suffering from EPs (diagnosed by hysteroscopy and histology), and group B included 240 patients without EPs at hysteroscopy. INTERVENTIONS: In group A, 2 separate samples were obtained from the EPs (group A polyps) and endometrium (group A endometrium). In group B, a single sample of endometrial tissue was evaluated (group B endometrium). All tissue samples were subjected to immunohistochemistry for CD-138 for plasma cell identification. MEASUREMENTS AND MAIN RESULTS: The primary study endpoint was to compare the rates of CE in group A endometrium versus group B endometrium. The secondary endpoint was to evaluate the consistency in CD-138 immunoreactivity between group A polyps and compared with group A endometrium. A higher prevalence of CE was observed in group A endometrium compared with group B endometrium (p < .0001). The total percentage of EPs showing CD-138 positivity was 76.7% (184 of 240). CE was more frequent in women with CD-138+ EPs compared to those with CD-138- EPs (p < .0001). CONCLUSIONS: EPs were commonly associated with CE in the premenopausal women suffering from AUB. Moreover, the majority of EPs were positive for CD-138 staining, suggesting a possible hidden association between chronic inflammation and EPs.

Office treatment of large endometrial polyps using truclear 5C: Feasibility and acceptability.

AIM: This retrospective multicenter study was carried out to evaluate feasibility, effectiveness and patient acceptability of a small diameter hysteroscopic tissue removal system in the treatment of large endometrial polyps (≥20 mm), usually not removed in an office setting. METHODS: Hundred and forty-six women with a single greater than 10-mm diameter polyp considered for polypectomy between April 2016 and August 2017. Sixty-five of these patients had a polyp size greater than 20 mm. All hysteroscopic polypectomy, using Hysteroscopic Tissue Removal system (TruClear 5C System Medtronic), were performed in an office setting with vaginoscopic approach. RESULTS: Procedural success, time to complete the polypectomy and patient pain scores were evaluated. Polyps less than 20 mm were completely removed in 79/81 cases (97.53%). The completeness of greater than or equal to 20-mm polyp removal was achieved in 63/65 cases (96.92%). The median time for polypectomy was 4.19 ± 1.03 min for polyps less than 20 mm and 4.97 ± 1.30 min for polyps greater than or equal to 20 mm, respectively. Pain was minimal and brief, and the mean pain score measured on a 10-point visual analog scale at the end of polypectomy showed no significant difference between the two groups. In 4/79 (5.06%) cases with polyps less than 20 mm and in 4/63 (6.35%) cases with polyps greater than or equal to 20 mm women reported moderate pain. All specimens were adequate for pathologic measurements. CONCLUSION: Hysteroscopic treatment of polyps greater than or equal to 20 mm in size with TruClear 5C is feasible and well tolerated in an office setting with no significant difference regarding completeness compared to polyps less than 20 mm, but with a minimal increase in procedure times.

Colpocytological abnormalities in HIV infected and uninfected pregnant women: prevalence, persistence and progression.

In this retrospective case-control study, we analyse data of 48 HIV-positive pregnant patients, versus a control group of 99 HIV-negative pregnant women, followed as outpatients by our department from 2009 to 2014. The aims of the study were to investigate the prevalence, persistence and progression of cervical squamous intraepithelial lesions (SIL) in each group and to correlate colpo-cytological lesions to the socio-demographic and clinical-laboratory findings in the HIV + pregnant women. In our study we observed that immunosuppression, HPV infection and vaginal coinfections were predictive of cervical lesions. Pap smear and colposcopy should be part of routine care for HIV-infected pregnant women because these lesions behave aggressively in these patients. Success of prevention depends on massive access of patients to screening. HAART reduces viral load and maintains CD4 count and can affect progression of SIL. Multidisciplinary services on the same site appear to be one promising strategy to improve compliance in patients. Impact Statement What is already known on this subject: Our study provided novel information on a highly vulnerable population of young HIV + pregnant women. What the results of this study add: We observed that immunosuppression, HPV infection and vaginal coinfections were predictive of cervical lesions remarkable with colposcopy. We could consider these important risk factors to evaluate to establish an appropriate strategy of management for these patients. What the implications are of these findings for clinical practice and/or further research: Association of the risk between SIL presence and HIV and HPV infection also deserves additional investigation. We believe that Pap smears and colposcopies should be part of the routine care for HIV-infected women because these lesions behave particularly aggressively in these patients.

Accuracy of Hysteroscopic Metroplasty With the Combination of Presurgical 3-Dimensional Ultrasonography and a Novel Graduated Intrauterine Palpator: A Randomized Controlled Trial.

UNLABELLED: STUDY OBJECTIVE: To assess whether the use of a novel graduated intrauterine palpator can improve the accuracy of hysteroscopic metroplasty, introducing objective intraoperative criteria. DESIGN: A prospective randomized study (Canadian Task Force I, evidence obtained from a properly design, randomized, controlled trial). SETTING: University Federico II hysteroscopic clinic. PATIENTS: Ninety women with a uterine septum diagnosed during office hysteroscopy and 3-dimesional transvaginal ultrasound (3D-TVS) were randomized into 2 groups: group T (metroplasty with intrauterine palpator) (n = 45) and group C (metroplasty without intrauterine palpator) (n = 45). INTERVENTIONS: Outpatient hysteroscopic metroplasty under conscious sedation using a 5-mm hysteroscope and miniaturized 5F instruments including a bipolar electrode for the removal of three quarters of the septum, blunt scissors to refine the base of the septum, and an intrauterine palpator to measure the portion of the removed septum (only group T). 3D-TVS and second-look hysteroscopy were used to identify the number of optimal (residual septum <5 mm), suboptimal (residual septum 5-10 mm), and incomplete resections (residual septum >10 mm). In group T, metroplasty was stopped when the intrauterine palpator showed that the resected septum corresponded to presurgical ultrasonographic measurements in order to obtain a fundal notch of 1.0 cm. In group C, metroplasty was interrupted once the tubal ostia were clearly visible on the same line and/or hemorrhage from small myometrial vessels of the fundus was observed. MEASUREMENTS AND MAIN RESULTS: No differences were observed in baseline characteristics between the 2 groups. The proportion of patients with complete septum resection was significantly higher in group T (71.5% vs 41%, χ(2): p = .006; relative risk: 1.684; 95% confidence interval, 1.116-2.506). Suboptimal resection was achieved in 13 cases (28.5%) in group T and 14 cases (20%) in group C, whereas incomplete resection was observed in only 12 patients in group C (27%). CONCLUSION: Presurgical evaluation with 3D-TVS together with the use of a graduate intrauterine palpator improves the accuracy of hysteroscopic metroplasty, allowing complete removal of a uterine septum in 1 surgical step.

Hysteroscopic Patterns in Women on Treatment With Ulipristal Acetate 5 mg/day: A Preliminary Study.

STUDY OBJECTIVE: To describe the hysteroscopic findings in women on treatment with ulipristal acetate (UPA) and to define the most common hysteroscopic patterns related to the treatment and compare them with the histologic findings. DESIGN: Preliminary study. SETTING: OB-GYN and Gynecology Oncology Clinic, Military Medical Institute, Ministry of Defense, Warsaw, Poland, and Obstetrics and Gynecology Department, University of Bari, Italy. PATIENTS: Seventy-four premenopausal patients complaining of abnormal uterine bleeding due to uterine myomas and on treatment with UPA 5 mg/day for at least 30 days. INTERVENTIONS: Women received transvaginal sonography (TVS) and then office hysteroscopy and visually guided endometrial biopsies. Video hysteroscopies were recorded in digital format. Pictures were evaluated by 2 authors off-line and compared with histologic results. MEASUREMENTS AND MAIN RESULTS: Hysteroscopic aspects and classification of progesterone receptor modulator-associated endometrial changes were measured. The most common hysteroscopic finding was the combination of a flat subtle epithelium with small glandular openings; large isolated or confluent cysts in the stroma, giving the surface a floating aspect at fluid distention; and well-evident subendometrial vascular network with a "chicken-wire" vascular pattern (44.6%). This finding accounted for 82% of cases with endometrial thickness > 10 mm at TVS. Histology confirmed a combination of epithelial secretory (vacuoles) and hypotrophic effects (small and dilated glands), whereas at stromal level the combination of cysts, dense stroma, and vascular wall thickening was found. At 3 months follow-up echographic, hysteroscopic, and histologic endometrial patterns were normal in all patients. CONCLUSIONS: In most women on UPA and with thickened endometrium at TVS, the hysteroscopy showed benign and characteristics aspects related to the ambivalent effects of UPA on progesterone receptor. These alterations took place just after 1 month of treatment but disappeared within 3 months of stopping treatment.

Morphokinetic parameters of early embryo development via time lapse monitoring and their effect on embryo selection and ICSI outcomes: a prospective cohort study.

PURPOSE: To compare the outcomes of embryos selected via time lapse monitoring (TLM) versus those selected with conventional methods of selection in subfertile women undergoing ICSI. METHODS: The study population (239 women) was classified into two groups, based on the monitoring method used: Group 1 (TLM) and Group 2 (conventional monitoring). Groups were compared according to the clinical and ICSI cycle characteristics and reproductive outcomes, while transfers were performed at day 2 or 3. Subgroup analyses were performed, in women of both groups according to age and clinical parameters, and in embryos of Group 1 based on their cellular events. RESULTS: There was a statistically significant difference between the two study groups with regard to the outcome parameters, favoring Group 1 and especially in women >40 years of age. No differences were found in subgroup analyses in participants of both groups, regarding the stimulation protocol used, number of the oocytes retrieved and type of subfertility, while in Group 1 the percentages of "in range" cellular events were higher in certain divisions in ages 35-40, non-smokers, and the GnRH-agonist group, and in embryos that resulted in pregnancy. CONCLUSION: Morphokinetic parameters of early embryo development via TLM are related to the characteristics of subfertile patients and associated with ICSI outcomes.

Statistical limits in sonographic estimation of birth weight.

PURPOSE: The accuracy of sonographic estimation of birth weight (EBW) is compromised by the precision of the biometrical measurements and the quality of the algorithms. This prospective study was to evaluate technical aspects to derive new equations for the EBW. METHODS: Three consecutive phases were carried out (1) to recruit a homogenous population, (2) to derive eight new algorithms using a multiple stepwise mathematical/statistical method, and (3) to test the accuracy of the developed equations. Only those patients with a singleton pregnancy who delivered within 48 h from the scan were considered for the analysis. RESULTS: The study population was made of 473 women. Four polynomial, two square root and two logarithmic algorithms were derived from a balanced study group of 200 women selected from the original study population. These formulas were subsequently applied and compared between them and showed a significant correlation with birth weight (p < 0.0001) and satisfactory statistical performances (r > 0.9), nevertheless they performed similarly to other equations previously published. CONCLUSIONS: The present findings define better the limitations associated with the intrinsic properties of algorithms and highlight that the possibility to improve the precision of sonographic measurements remains the only point at issue to increase the accuracy in the prediction of birth weight.

Estimation of birthweight by measurement of fetal thigh soft-tissue thickness improves the detection of macrosomic fetuses.

The accuracy of sonographic estimation of birthweight in suspected macrosomic fetuses is compromised by the imprecision of the biometrical measurements. This prospective study evaluated the performance of an equation based on linear measurement of the soft tissue above the external side of the fetal femur. The performance of this algorithm was compared with two classical algorithms. Sonographic measurements were taken within 48 h before of delivery. Sixty-two patients with fetuses with suspected macrosomia were enrolled. The studied formulas were compared between them and showed a significant correlation with birthweight (p < 0.0001) and satisfactory statistical performances (r > 0.9). The new formula showed a reduced standard deviation that means a lower internal error in the prediction. This study supports the potential of this new approach for the estimation of birthweight in large fetuses based on sonographic linear measurements only.