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Primary high-grade serous cancer arising from uterosacral ligament endometriosis: two case reports.

ABSTRACT: Although high-grade serous cancer (HGSC) accounts for >70% of ovarian epithelial cancers, it is rarely associated with endometriosis. No previous study has reported an association between the malignant transformation of uterine ligament endometriosis and HGSC. Here, we reported two cases of Chinese female patients with HGSC arising from endometriosis in the uterosacral ligament. They had a long-term history of endometriosis and dysmenorrhea. Both were diagnosed with HGSC at stage IIB. They underwent operations and six cycles of chemotherapy with paclitaxel and carboplatin and have remained disease-free to date. Genomic analysis showed no known/suspected pathogenic variations or somatic homologous recombination deficiency in the two cases. In conclusion, these rare cases of HGSC from endometriosis might indicate a new origin of ovarian type II carcinoma. Patients with a long-term history of endometriosis and sudden aggravation of dysmenorrhea or vaginal bleeding should be aware of the possibility of endometriotic malignant transformation.

SUBMITTER: Chen M

PROVIDER: S-EPMC9290122 | biostudies-literature | 2022 Jul

REPOSITORIES: biostudies-literature

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Primary high-grade serous cancer arising from uterosacral ligament endometriosis: two case reports.

Chen Ming M Zhang Yuedi Y Xu Manman M Liu Duo D Yang Yang Y Yao Shuzhong S

The Journal of international medical research 20220701 7

Although high-grade serous cancer (HGSC) accounts for >70% of ovarian epithelial cancers, it is rarely associated with endometriosis. No previous study has reported an association between the malignant transformation of uterine ligament endometriosis and HGSC. Here, we reported two cases of Chinese female patients with HGSC arising from endometriosis in the uterosacral ligament. They had a long-term history of endometriosis and dysmenorrhea. Both were diagnosed with HGSC at stage IIB. They under ...[more]

PMID: 35836383

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Project description:IntroductionLaparoscopic sacral colpopexy is the gold standard technique for apical prolapse correction but it is a technically challenging procedure with rare but severe morbidity. Laparoscopic high uterosacral ligament suspension could be a valid technically easier alternative using native tissue.Material and methodsIn the period from 2015 to 2018, 600 women were submitted to laparoscopic sacral colpopexy while 150 to laparoscopic high uterosacral ligament suspension in three Italian urogynecology referral centers. We enrolled women with apical prolapse stage ≥2 alone or multicompartment descensus. To reduce allocation bias, we performed a propensity matched analysis. Women undergoing laparoscopic high uterosacral ligament suspension surgery were matched 1:2 to women undergoing laparoscopic sacral colpopexy. The cumulative proportion of relapse-free women in time was analyzed by the Kaplan-Meier method. The primary objective of this multicenter case-control retrospective study was to compare the recurrence rate while the secondary objectives were to compare feasibility, safety, and efficacy of laparoscopic sacral colpopexy and laparoscopic high uterosacral ligament suspension in surgical treatment of pelvic organ prolapse.ResultsThree hundred and nine women were enrolled (103 laparoscopic high uterosacral ligament suspension; 206 laparoscopic sacral colpopexy). Median operatory time was significantly shorter in the laparoscopic high uterosacral ligament suspension group (P = 0.0001). No statistically significative difference was found in terms of estimated blood loss, admission time, intraoperative, and major early postoperative complications, postoperative pelvic pain, dyspareunia and de novo stress urinary incontinence. Surgical approach was the only independent risk factor for prolapse recurrence (RR = 6.013 [2.965-12.193], P = 0.0001). The objective cure rate was higher in the laparoscopic sacral colpopexy group (93.7 vs. 68%, 193/206 vs. 70/103, P = 0.0001) with a highly reduced risk of recurrence (RR = 5.430 [1.660-17.765]). Median follow up was 22 months.ConclusionBoth techniques are safe, feasible, and effective. Laparoscopic sacral colpopexy remains the best choice in treatment of multicompartment and advanced pelvic organ prolapse while laparoscopic high uterosacral ligament suspension could be appropriate for moderate and isolated apical prolapse when laparoscopic sacral colpopexy is not suitable for the patient or to prevent prolapse in women at high risk at the time of the hysterectomy.

Project description:Primary carnitine deficiency (PCD) is a disorder of the carnitine cycle that results in defective fatty acid oxidation. When carnitine cannot be transported into the cells, fatty acid oxidation is impaired, resulting a variety of symptoms, such as chronic muscle weakness, cardiomyopathy, hypoglycemia and liver dysfunction. The clinical manifestations and outcomes of different cases with PCD vary among patients. The present case report focused on two sisters with PCD. The younger sister presented with intractable epilepsy, and the older sister presented with reversible metabolic cardiomyopathy. Potential mutations in the SLC22A5 gene were investigated within the family, and a nonsense mutation [c.760C>T (p.R254X)] was identified in four family members. The two sisters harbored homozygous mutations, whereas their parents presented heterozygous mutations. Metabolic disease screening revealed low plasma free carnitine levels (<5 µmol/l) in the two sisters. The plasma free carnitine levels of their parents were normal, and they were asymptomatic. PCD in the two patients was managed using oral levocarnitine. The metabolic cardiomyopathy of the older sister improved following 3 months of treatment. However, the epilepsy of the younger sister was recurrent with oral antiepileptic therapy lasting one year and eight months, and epilepsy was finally controlled following right cerebral resection. The present case report demonstrated that the clinical manifestations presented by patients with PCD within the same family were different. The results indicated that treatment with levocarnitine supplementation should be initiated as soon as possible before irreversible organ damage occurs. In addition, metabolic decompensation and cardiac muscle functions were improved following carnitine supplementation. The resection of the severely diseased unilateral brain combined with carnitine supplementation and antiepileptic therapy may be an effective treatment for PCD with intractable epilepsy complications.

Dataset Information

Primary high-grade serous cancer arising from uterosacral ligament endometriosis: two case reports.

Publications

Primary high-grade serous cancer arising from uterosacral ligament endometriosis: two case reports.

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