Lymphangioleiomyomatosis (LAM) is a rare disease that typically affects women of childbearing age. It most commonly affects the lungs (P-LAM) but can occasionally occur in extra-pulmonary sites (E-LAM). There is a strong association between LAM and the tuberous sclerosis complex (TSC). We report a case of a 42-year-old female TSC sufferer who presented with dysfunctional uterine bleeding. She was not known to have LAM. An endometrial biopsy revealed a spindled-cell lesion suspicious of leiomyosarcoma, which correlated with cross-sectional imaging. She underwent a hysterectomy that showed a bizarre (symplastic) leiomyomatous endometrial polyp with background uterine LAM. We discuss the clinical and pathological implications of this unusual case of E-LAM and the importance of clinicopathological correlation in TSC sufferers. The association of uterine LAM with TSC is important and LAM should be considered as a differential of dysfunctional uterine bleeding and a benign mimic to uterine leiomyosarcoma in patients with TSC.
This study provides useful insight into the impact of extensive surgery on patients. Further multicenter studies are needed to evaluate the impact of extensive surgery on patient's QoL and survival.
Aim: Cervical stenosis is traditionally managed by mechanical dilatation under general anesthesia (GA). We aimed to assess the safety, effectiveness, and patient acceptability of dilatation in the outpatient setting under local anesthesia (LA). Methods: Data were collected prospectively from all patients attending the outpatient department with cervical stenosis from March 20, 2015 to September 23, 2020. Mechanical dilatation of the cervix was performed using Hegar dilators under LA. Subsequent colposcopic assessment, cytology, histology, and management were recorded. Results: One hundred forty-nine cases were referred for cervical dilatation, 63 (43%) of which had complete stenosis. One hundred eighteen (79%) patients had previously undergone cervical procedures. Successful dilatation under LA was achieved in 119 (83%) patients; 5 (3%) declined (requesting GA), 6 (4%) did not tolerate speculum examination, and 19 (13%) had unsuccessful procedures. The median Hegar size used was 8 mm. Dilatation under LA was acceptable in 93% attempted procedures. Thirteen episodes of restenosis were recorded with no major adverse events. Younger age (p = 0.045) and severe (compared to complete) stenosis (p < 0.0001) were associated with procedure success, with improved results over time (p = 0.003). Successful dilatation permitted cervical assessment; eight patients required cervical excisions, two underwent hysterectomies, with one confirmed case of adenocarcinoma. Conclusion: Rigid cervical dilatation in the outpatient setting provides effective, instantaneous treatment for women who have failed cytological or colposcopic assessment. For the vast majority of women, the procedure was well tolerated and preferred to using GA. However, given that 1 in 10 women experienced restenosis, patients should be counseled about the possibility of requiring further management.
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