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Asian Journal of Surgery Feb 2024
Topics: Female; Humans; Brenner Tumor; Ovarian Neoplasms; Pelvis; Cysts
PubMed: 37981498
DOI: 10.1016/j.asjsur.2023.11.033 -
FEBS Open Bio Jan 2024Male and female reproductive tracts develop from anterior intermediate mesoderm with similar differentiation processes. The anterior intermediate mesoderm develops into...
Male and female reproductive tracts develop from anterior intermediate mesoderm with similar differentiation processes. The anterior intermediate mesoderm develops into the mesonephros, and the Wolffian duct initiates by epithelialization in the mesonephros. The Müllerian duct invaginates from the coelomic epithelium of the cranial mesonephros for ductal formation and is then regionalized into proximal to caudal female reproductive tracts. In this study, we focused on the epithelialization of the Wolffian duct, initiation of the Müllerian duct, and the regionalization step of the Müllerian ducts as a continuous process. By using intermediate mesodermal cells from mouse pluripotent stem cells, we identified that inhibition of SMAD2/3 signaling might be involved in the differentiation into mesenchymal cells, after which mesonephric cells might be then epithelialized during differentiation of the Wolffian duct. Aggregation of coelomic epithelial cells might be related to initiation of the Müllerian duct. Transcriptomic analysis predicted that consensus sequences of SMAD3/4 were enriched among highly expressed genes in the proximal Müllerian duct. SMAD2/3 signaling to regulate differentiation of the Wolffian duct was continuously activated in the proximal Müllerian duct and was involved in proximal and oviductal regionalization. Therefore, SMAD2/3 signaling may be finely tuned to regulate differentiation from initiation to regionalization steps.
Topics: Mice; Animals; Male; Female; Wolffian Ducts; Mullerian Ducts; Cell Differentiation; Epithelial Cells; Signal Transduction
PubMed: 37953493
DOI: 10.1002/2211-5463.13729 -
Radiology Case Reports Dec 2023Herlyn-Werner-Wunderlich (HWW) syndrome is a rare anomaly of the female urogenital tract characterized by the combination of uterine didelphys, obstructed hemivagina,...
Herlyn-Werner-Wunderlich (HWW) syndrome is a rare anomaly of the female urogenital tract characterized by the combination of uterine didelphys, obstructed hemivagina, and ipsilateral renal anomalies. The exact incidence of the syndrome remains unknown, but it has been reported to be 1 per 2000 to 1 per 28,000 women. It is believed that the triad is a mesonephric duct-induced paramesonephric duct anomaly. In majority of the cases with complete hemivaginal obstruction, the pathology is diagnosed after menarche due to retained menstrual flow. While the common clinical presentations are dysmenorrhea, pelvic pain, intermenstrual bleeding, and pelvic mass, it can also manifest itself with unusual gastrointestinal and urinary tract symptoms. We present a case of HWW syndrome with gastrointestinal symptoms like worsening constipation and abdominal fullness. The unusual clinical presentation of this syndrome makes diagnosis more challenging. To solve such medical puzzles and prevent complications, detailed history-taking and radiological guidance are critical.
PubMed: 37929046
DOI: 10.1016/j.radcr.2023.08.117 -
Indian Journal of Nephrology 2023Mesonephric remnants persist as an appendix of epididymis and paradidymis in efferent ductules in males and skene's glands and Gartner's ducts in females. The...
Mesonephric remnants persist as an appendix of epididymis and paradidymis in efferent ductules in males and skene's glands and Gartner's ducts in females. The mesonephric remnant in the renal parenchyma is extremely rare and only a few cases have been reported in the literature. We present a case with a non-functioning atrophic left kidney. Histopathology showed variable-sized ducts filled with colloid-like material surrounded by collagenized stroma. The ureter showed hypertrophied muscle and a few ducts lined by flattened and a few by columnar epithelium resembling epididymis suggestive of mesonephric remnants. IHC for CD10, PAX 8, and GATA3 was positive. A diagnosis of congenital unilateral hypoplasia of kidneys and ureter with mesonephric remnants was given.
PubMed: 37881746
DOI: 10.4103/ijn.IJN_579_20 -
CRSLS : MIS Case Reports From SLS 2023Endometriosis originating in mesonephric cyst is unusual and with unknown prevalence. Endometriotic lesion in vestigial remnant of wolffian duct (mesonephric cyst) is... (Review)
Review
OBJECTIVES
Endometriosis originating in mesonephric cyst is unusual and with unknown prevalence. Endometriotic lesion in vestigial remnant of wolffian duct (mesonephric cyst) is exceptional. In the extended literature review only three cases have been reported in animal studies, and our case reported here is the first in human beings. We present a case of mesonephric cyst endometrioma in a 37-year-old patient who was referred for severe dysmenorrhea, long duration pelvic and back pain, subfertility, severe dyspareunia, and groin discomfort. The patient underwent laparoscopic removal and we performed a literature review to gain insight about the origin and surgical management of an atypical site endometriosis.
METHODS AND PROCEDURES
Case report presentation rests on information obtained from the patient database. We performed the literature review using a Medline search with the keywords: mesonephric cyst endometriosis, atypical location of endometriosis in vestigial remnant in wolffian duct, and Gartner duct cyst endometrioma.
RESULTS
On physical examination, fullness and tenderness in left adnexa and lateral vaginal wall fullness on left side with restricted mobility of uterus was noted. Based on the examination and imaging the left ovarian cyst and mesonephric cyst were suspected. Surgical exploration revealed the left hemorrhagic cyst with deep infiltrating endometriosis involving left ureter and left uterosacral ligament with mesonephric cyst endometriosis. The review of literature revealed three cases where ectopic endometrial tissue in mesonephric cyst remnant was found in female dogs.
CONCLUSION
Mesonephric cyst endometrioma, although rare, can be a representative of extensive endometriosis. This case highlights an importance of careful clinical examination, correlation of patient symptoms with examination and imaging, and successful laparoscopic management of an atypical location endometriotic lesions. We completed the literature review on successful surgical management of such cases.
Topics: Animals; Dogs; Female; Humans; Adult; Endometriosis; Laparoscopy; Ovarian Cysts; Pelvis; Cysts
PubMed: 37808583
DOI: 10.4293/CRSLS.2023.00029 -
SA Journal of Radiology 2023Tubulocystic anomalies of the mesonephric duct (MND) are a rare group of related entities with a perplexing clinical presentation. Ultrasound is a useful screening...
UNLABELLED
Tubulocystic anomalies of the mesonephric duct (MND) are a rare group of related entities with a perplexing clinical presentation. Ultrasound is a useful screening investigation, which can help identify a dysplastic kidney or point to renal agenesis and identify cystic or tubulocystic changes in the structures derived from the ureteric bud or MND. Further evaluation with MRI can help in characterisation and direct management. The presence of a dysplastic kidney or absence of a kidney should prompt further evaluation for associated ureteric abnormalities such as ectopic insertion or ureterocoele, which could lead to a diagnosis of tubulocystic anomaly of the MND.
CONTRIBUTION
The authors describe two cases, briefly outline the diagnostic approach and summarise the literature on management. Reporting radiologists should be aware of these entities.
PubMed: 37794947
DOI: 10.4102/sajr.v27i1.2700 -
Asian Journal of Surgery Dec 2023
Topics: Female; Humans; Adenocarcinoma; Endometrium
PubMed: 37730504
DOI: 10.1016/j.asjsur.2023.09.029 -
Journal of Cancer Research and Clinical... Nov 2023Mesonephric-like adenocarcinomas (MLA) of the female genital tract represent a rare and relatively recently described neoplasm exhibiting characteristic morphologic and... (Review)
Review
Mesonephric-like adenocarcinoma of the female genital tract: possible role of KRAS-targeted treatment-detailed molecular analysis of a case series and review of the literature for targetable somatic KRAS-mutations.
PURPOSE
Mesonephric-like adenocarcinomas (MLA) of the female genital tract represent a rare and relatively recently described neoplasm exhibiting characteristic morphologic and immunohistochemical findings commonly associated with a KRAS-mutation. Most cases display an aggressive clinical behavior, but knowledge about treatment approaches is limited, especially for targeting KRAS.
METHODS
We report a series of eight cases with a detailed molecular analysis for KRAS. These cases as well as the data of previously published cases with detailed information regarding KRAS-mutational events were reviewed for a potential targeted approach and its prognostic impact.
RESULTS
Both the uterine and ovarian MLA harbor a somatic KRAS-mutation in about 85% of the reported cases, affecting the hotspot codons 12 and 13. 15.7% of the endometrial and 15.6% of ovarian MLA are wild type for KRAS. A p.G12A-alteration was seen in 5.6% (5/89) of the endometrial and in 6.2% (2/32) of the ovarian tumors, for p.G12C in 7.9% and 6.2%, for p.G12D in 32.6% and 34.5% and for p.G12V in 36% and 37.5%, respectively. Very limited data are available regarding the prognostic impact of different mutational sites within the KRAS-gene without significant prognostic impact.
CONCLUSION
Because of a specific p.G12C-KRAS somatic mutation, only the minority of MLA (7.9% with uterine and 6.2% with ovarian primary) are potentially targetable by sotarasib in that rare but aggressive subtype of adenocarcinoma of the female genital tract. Until now, the different location of a somatic KRAS-mutation is of no prognostic impact.
Topics: Humans; Female; Proto-Oncogene Proteins p21(ras); Adenocarcinoma; Mutation; Prognosis; Genitalia, Female
PubMed: 37668797
DOI: 10.1007/s00432-023-05306-9 -
Biomedicines Aug 2023Data on genetic and immunophenotypical characteristics of uterine mesonephric-like adenocarcinoma (MLA) remain limited. Therefore, we aimed to investigate the...
Mesonephric-like Adenocarcinoma of the Uterine Corpus: Genomic and Immunohistochemical Profiling with Comprehensive Clinicopathological Analysis of 17 Consecutive Cases from a Single Institution.
Data on genetic and immunophenotypical characteristics of uterine mesonephric-like adenocarcinoma (MLA) remain limited. Therefore, we aimed to investigate the clinicopathological, immunohistochemical, and molecular features of uterine MLA. We performed targeted sequencing, array comparative genomic hybridization, and immunostaining in 17, 13, and 17 uterine MLA cases, respectively. Nine patients developed lung metastases. Eleven patients experienced disease recurrences. The most frequently mutated gene was Kirsten rat sarcoma viral oncogene homolog (; 13/17). Both the primary and matched metastatic tumors harbored identical (3/4) and phosphatase and tensin homolog deleted on chromosome 10 (1/4) mutations, and did not harbor any additional mutations. A total of 2 of the 17 cases harbored tumor protein 53 () frameshift insertion and deletion, respectively. Chromosomal gains were detected in 1q (13/13), 10 (13/13), 20 (10/13), 2 (9/13), and 12 (6/13). Programmed cell death-ligand 1 overexpression or mismatch repair deficiency was not observed in any of the cases. Initial serosal extension and lung metastasis independently predicted recurrence-free survival with hazard ratios of 6.30 and 7.31, respectively. Our observations consolidated the clinicopathological and molecular characteristics of uterine MLA. Both clinicians and pathologists should consider these features to make an accurate diagnosis of uterine MLA and to ensure appropriate therapeutic management of this rare entity.
PubMed: 37626765
DOI: 10.3390/biomedicines11082269 -
International Journal of Gynecological... May 2024Immunohistochemical markers shown to be useful in identifying/confirming mesonephric/mesonephric-like differentiation (MLD markers) include thyroid transcription factor...
Immunohistochemical markers shown to be useful in identifying/confirming mesonephric/mesonephric-like differentiation (MLD markers) include thyroid transcription factor (TTF1), GATA-binding protein 3 (GATA3), and cluster of differentiation 10 (CD10). Only a few studies have examined the expression levels of MLD markers in endometrial endometrioid carcinomas (EECs). This study aimed to analyze the frequency and pattern of MLD marker expression in low-grade EECs. We performed immunostaining for the detection of TTF1, GATA3, and CD10 expression in 50 low-grade EEC tissue samples and evaluated their staining proportion and intensity. Nine tumors (18.0%) expressed at least one MLD marker in varying proportions and intensities, and 2 of these tumors were positive for 2 MLD markers (TTF1/GATA3 and GATA3/CD10, respectively). Three (6.0%) tumors showed moderate-to-strong nuclear TTF1 immunoreactivity in ≤5% of the tumor cells. Five tumors (10.0%) had at least moderate nuclear GATA3 staining, and three of them displayed a staining proportion of ≥15%. Three tumors (6.0%) were focal (mean proportion, 15%) but strongly positive for CD10. Our findings indicate that a subset of EEC can express one or more MLD markers with varying staining proportions and intensities. Given that a diagnosis of uterine mesonephric-like adenocarcinoma should be established based on a combination of characteristic histologic features, unique immunophenotypes, and confirmed molecular findings, pathologists should not exclude EEC based only on the presence of focal immunoreactivity for MLD markers. Awareness of the atypical expression patterns of MLD markers in EEC helps pathologists avoid misdiagnosing EEC as a uterine mesonephric-like adenocarcinoma.
Topics: Female; Humans; Carcinoma, Endometrioid; Mesonephros; Uterus; Adenocarcinoma; Biomarkers, Tumor; Endometrial Neoplasms
PubMed: 37566876
DOI: 10.1097/PGP.0000000000000976