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Medicine Nov 2020Mesonephric-like adenocarcinoma (MLA) from ovary is a very rare tumor which derives from mesonephric duct remnant of the female genital tract. Only six cases have been... (Review)
Review
RATIONALE
Mesonephric-like adenocarcinoma (MLA) from ovary is a very rare tumor which derives from mesonephric duct remnant of the female genital tract. Only six cases have been reported so far in the English literature.
PATIENT CONCERNS
A 29-year-old female patient was referred to the local hospital with a 20-day history of abdominal discomfort.
DIAGNOSES
Pelvic ultrasound examination revealed a solid and cystic mass measuring 10 cm in diameter in the right adnexal area and a cystic mass measuring 5 cm in the left adnexal area. Postoperative pathology in the local hospital revealed suspected malignancy of the right ovary, and she was then transferred to our institution for definite diagnosis. The tumor mass was finally diagnosed as a primary MLA arising from the right ovary by histological and immunohistochemical examination in our institution.
INTERVENTIONS
The patient underwent laparoscopic right adnexectomy and removal of left ovarian cyst in the local institution. Then, she underwent a complete staging surgery including a total hysterectomy, left adnexectomy, pelvic plus para-aortic lymphadenectomy, and omentectomy in our hospital. In addition, she received four cycles of combination chemotherapy with carboplatin plus paclitaxel.
OUTCOMES
There is no evidence of recurrence with 13 months of follow-up till now, and we are still following-up this patient.
LESSONS
MLA is an extremely uncommon malignancy with difficult diagnosis, unclear treatment and poor prognosis. Familiarizing with the clinical features and optimal management of this rare tumor may increase awareness of the disease among clinicians and pathologists, thus avoiding the misdiagnosis and mistreatment.
Topics: Adenocarcinoma; Adult; Chemotherapy, Adjuvant; Female; Humans; Ovarian Neoplasms; Rare Diseases; Wolffian Ducts
PubMed: 33235131
DOI: 10.1097/MD.0000000000023450 -
Asian Journal of Surgery Dec 2023
Topics: Female; Humans; Adenocarcinoma; Endometrium
PubMed: 37730504
DOI: 10.1016/j.asjsur.2023.09.029 -
Modern Pathology : An Official Journal... Mar 2004The diagnosis of prostatic adenocarcinoma, especially when present in small amounts, is often challenging. Before making a diagnosis of carcinoma, it is prudent for the... (Review)
Review
The diagnosis of prostatic adenocarcinoma, especially when present in small amounts, is often challenging. Before making a diagnosis of carcinoma, it is prudent for the pathologist to consider the various benign patterns and processes that can simulate prostatic adenocarcinoma. A useful method of classifying benign mimickers is in relationship to the major growth patterns depicted in the classical Gleason diagram. The four major patterns are small gland, large gland, fused gland and solid. Most mimickers fit within the small gland category and the most common ones giving rise to false-positive cancer diagnosis are atrophy, post-atrophic hyperplasia, atypical adenomatous hyperplasia and seminal vesicle-type tissue. A number of other histoanatomic structures such as Cowper's gland, verumontanum mucosal glands, mesonephric glands and paraganglionic tissue may be confused with adenocarcinoma. Additionally, metaplastic and hyperplastic processes within the prostate may be confused with adenocarcinoma. Furthermore, inflammatory processes including granulomatous prostatitis, xanthogranulomatous prostatitis and malakoplakia may simulate high-grade adenocarcinoma. Atypical adenomatous hyperplasia (adenosis), a putative precursor of transition zone adenocarcinoma, has overlapping features with low-grade adenocarcinoma and may cause problems in differential diagnosis, especially in the needle biopsy setting. The pathologist's awareness of the vast array of benign mimickers is important in the systematic approach to the diagnosis of prostatic adenocarcinoma. Knowledge of these patterns on routine microscopy coupled with the prudent use of immunohistochemistry will lead to a correct diagnosis and avert a false-positive cancer interpretation.
Topics: Adenocarcinoma; Diagnosis, Differential; Humans; Male; Prostate; Prostatic Diseases; Prostatic Neoplasms
PubMed: 14976539
DOI: 10.1038/modpathol.3800055 -
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 -
Cells Apr 2021Mixed epithelial and stromal tumor of the kidney (MESTK), a benign rare tumor with malignant transformation potential, is thought to be derived from fetal or immature...
Mixed epithelial and stromal tumor of the kidney (MESTK), a benign rare tumor with malignant transformation potential, is thought to be derived from fetal or immature cells originating from the mesonephric and Müllerian ducts. However, due to its rarity, little is known about the anti-tumor immune responses in MESTK. Herein, we present five cases of MESTK and evaluate the population of tumor-infiltrating lymphocytes (TILs) using a freshly obtained MESTK sample. Microscopically, TILs were scattered or clustered in large aggregates in the stroma in all five cases; furthermore, three cases exhibited heavy, large lymphocytic aggregates with no well-organized tertiary lymphoid structures with germinal centers. Flow cytometric analysis of TILs in one freshly obtained MESTK sample revealed that >40% of CD3 T cells were effector memory FasCD28 γδ T cells expressing high levels of programmed cell death protein 1 and inducible T-cell co-stimulator, but low levels of CD44 and CD27. Most αß T cells exhibited a naïve phenotype. Additionally, we detected many activated class-switched CD21CD27 B cells as well as CD11cIgM marginal zone B-like and CD27CD21CD23 immunoglobulin (Ig)DIgM age-associated B-like cells. Collectively, for the first time, we report the immune microenvironment pattern of MESTK to oncogenic stress.
Topics: Adult; Epithelial Cells; Female; Humans; Kidney Neoplasms; Lymphocytes, Tumor-Infiltrating; Male; Middle Aged; Stromal Cells; Tomography, X-Ray Computed
PubMed: 33923583
DOI: 10.3390/cells10040917 -
Journal of Pediatric Hematology/oncology May 2022Primary extrarenal Wilms tumors are rare neoplasms that are presumed to arise from metanephric or mesonephric remnants outside of the kidney. Their pathogenesis is...
Primary extrarenal Wilms tumors are rare neoplasms that are presumed to arise from metanephric or mesonephric remnants outside of the kidney. Their pathogenesis is debated but has not been studied, and there are no reports of genomic descriptions of extrarenal Wilms tumors. We describe a diffusely anaplastic extrarenal Wilms tumor that occurred in the lower abdomen and upper pelvis of a 10-year-old boy. In addition to the clinical, histopathologic, and radiologic features, we describe the cytogenetic changes and exomic profile of the tumor. The tumor showed loss of the tumor suppressor AMER1, loss of chromosome regions 1p, 16q, and 22q, gain of chromosome 8, and loss of function TP53 mutation-findings known to occur in renal Wilms tumors. This is the first description of the exomic profile of a primary extrarenal Wilms tumor. Our data indicate that primary extrarenal Wilms tumors may follow the same pathogenetic pathways that are seen in renal Wilms tumors. Finally, we describe the establishment of first ever tumor models (primary cell line and patient-derived xenograft) from an extrarenal Wilms tumor.
Topics: Child; Female; Humans; Kidney; Kidney Neoplasms; Male; Mutation; Wilms Tumor
PubMed: 35129140
DOI: 10.1097/MPH.0000000000002413 -
Gynecologic Oncology Reports Nov 2020Mesonephric carcinoma is a rare cancer that most often arises within the cervix, and less frequently, in the ovary and endometrium. A retrospective search of our...
Mesonephric carcinoma is a rare cancer that most often arises within the cervix, and less frequently, in the ovary and endometrium. A retrospective search of our CLIA-certified and CAP-accredited reference molecular laboratory database (Foundation Medicine, Inc.) identified 20 mesonephric or mesonephric-like, cervical (n = 10), endometrial (n = 5), ovarian (n = 4) or peri-bladder (n = 1) carcinomas that had undergone comprehensive genomic profiling via next generation sequencing. Activating mutations were present in 90%, 18 of 20 cases, including G12V (n = 7), G12D (n = 6), G12A (n = 3) and G12C (n = 2). Other recurrent alterations were identified in (25%), (20%), (15%), (10%), (10%) and (10%). One wild-type case had a mutation as the sole alteration, while the second wild-type case had an exon 20 insertion D770_N771insSVD alteration. All tumors were negative for HPV DNA, microsatellite instability, high tumor mutational burden and homologous recombination deficiency. A circulating tumor DNA (ctDNA) liquid biopsy from peripheral blood, which was performed 6 years after original solid tumor resection in one patient with suspected lung metastasis, revealed concordance of alteration, gains of chromosomes 1q, 2, 10, 12 and 20, plus new alterations in the liquid biopsy compared to the original sample. G12 mutation is major driver of mesonephric and mesonephric-like carcinomas, with less frequent contribution by ARID1A and PIK3CA pathways in tumors of non-cervical origin. ctDNA liquid biopsy may be useful in detecting mutations in recurrent or metastatic patients, who may potentially be eligible for trials against emerging targeted therapies.
PubMed: 33024807
DOI: 10.1016/j.gore.2020.100652 -
Diagnostics (Basel, Switzerland) Nov 2021Mesonephric-like adenocarcinoma (MLA) of the uterine corpus is a rare but distinct malignant tumor of the female genital tract, demonstrating a characteristic morphology...
Mesonephric-like adenocarcinoma (MLA) of the uterine corpus is a rare but distinct malignant tumor of the female genital tract, demonstrating a characteristic morphology and unique immunohistochemical profiles and molecular alterations. We conducted immunohistochemical staining (IHC) to make precise differential diagnoses of uterine MLAs from common histological subtypes of endometrial carcinomas. We collected 25 uterine MLAs and performed IHC for GATA3, TTF1, CD10, ER, PR, p16, p53, and HER2. Seventeen cases (68.0%) showed at least moderate nuclear GATA3 immunoreactivity in ≥25% of tumor cells. Most cases expressed TTF1 (17/21, 81.0%) and CD10 (luminal; 17/21, 81.0%). Heterogeneous TTF1 expression was noted in 12 cases. An inverse pattern of GATA3 and TTF1 staining was observed in eight cases (32.0%). Three cases (12.0%) showed moderate-to-strong ER expression in ≥25% of tumor cells, and two cases (8.0%) showed moderate-to-strong PR expression in ≥5% of tumor cells. These hormone receptor-positive MLAs varied in intensity and proportion of GATA3 staining. None of the 25 cases exhibited either diffuse and strong p16 expression or aberrant p53 expression. Five cases (20.0%) showed equivocal HER2 immunoreactivity (score 2+), but FISH confirmed that none of them exhibited gene amplification. In summary, a small subset of uterine MLAs displayed atypical IHC results: focal but strong expression of ER or PR, the complete absence of GATA3 immunoreactivity, the concurrent expression of mesonephric and hormone receptors, and the inverse pattern of GATA3 and TTF1 staining. These unusual immunophenotypes may complicate the differential diagnosis of MLA. Moreover, pathologists should be encouraged to interpret the IHC results cautiously.
PubMed: 34829389
DOI: 10.3390/diagnostics11112042 -
Diagnostics (Basel, Switzerland) Jan 2022Mesonephric adenocarcinoma (MA) of the female genital tract is a rare but distinct entity, exhibiting unique morphological, immunophenotypical, and molecular...
Mesonephric adenocarcinoma (MA) of the female genital tract is a rare but distinct entity, exhibiting unique morphological, immunophenotypical, and molecular characteristics. Vaginal MA is hypothesized to arise from the mesonephric remnants located in the lateral vaginal wall. A 52-year-old woman presented with vaginal bleeding. Physical examination revealed a protruding mass in the left vaginal wall. Pelvic magnetic resonance imaging revealed a 2.5-cm mass arising from the left upper vagina and extending posterolaterally to the extravaginal tissue. The punch biopsy was diagnosed as poorly differentiated adenocarcinoma. She received radical surgical resection. Histologically, the tumor displayed various architectural patterns, including compactly aggregated small tubules, solid cellular sheets, endometrioid-like glands and ducts, intraluminal micropapillae, cribriform structure, and small angulated glands accompanied by prominent desmoplastic stroma. The tubules and ducts possessed hyaline-like, densely eosinophilic intraluminal secretions. The tumor extended to the subvaginal soft tissue and had substantial perineural invasion. Immunostaining revealed positivity for the mesonephric markers, including GATA3, TTF1, and PAX2, while showing very focal and weak positivity for estrogen receptor and negativity for progesterone receptor. Additionally, we observed a complete absence of p53 immunoreactivity. Targeted sequencing analysis revealed that the tumor harbored both activating p.G12D mutation and truncating p.E286* mutation. A thorough review of the previous literature revealed that 4.5% (3/67) of vaginal/cervical MAs and 0.9% (1/112) of uterine/ovarian mesonephric-like adenocarcinomas harbor mutations, indicating that this is very uncommon in malignant mesonephric lesions. In summary, we presented a rare case of vaginal MA uniquely harboring pathogenic mutation, resulting in p53 aberration.
PubMed: 35054285
DOI: 10.3390/diagnostics12010119 -
Cancer Genomics & Proteomics 2022Uterine mesonephric-like adenocarcinoma (MLA) is a rare malignant tumor of the female genital tract. (Review)
Review
Mesonephric-like Adenocarcinoma of the Uterine Corpus: Comprehensive Analyses of Clinicopathological, Molecular, and Prognostic Characteristics With Retrospective Review of 237 Endometrial Carcinoma Cases.
BACKGROUND/AIM
Uterine mesonephric-like adenocarcinoma (MLA) is a rare malignant tumor of the female genital tract.
PATIENTS AND METHODS
We reviewed 237 endometrial carcinoma cases and investigated the clinicopathological and molecular characteristics of uterine MLA.
RESULTS
We found that 3.0% (7/237) of the endometrial carcinoma cases were MLAs. Compared to endometrial endometrioid carcinoma, MLA showed larger tumor size, deeper myometrial invasion, increasingly advanced-stage disease, and more frequent lymphovascular space invasion. All MLAs exhibited architectural diversity, compactly aggregated small tubules, eosinophilic intraluminal secretions, overlapped and angulated nuclei, scant cytoplasm, and presence of spindle cells. All the MLAs expressed at least two mesonephric markers. All except one MLA harbored activating Kirsten rat sarcoma viral oncogene homolog mutations. All patients with MLA developed postoperative metastases. MLA had the lowest progression-free survival rate among different histological types of endometrial carcinoma.
CONCLUSION
Uterine MLA is a highly aggressive gynecological malignancy, showing unique morphological and molecular features, frequent recurrences and metastases, as well as poor prognosis.
Topics: Adenocarcinoma; Carcinoma, Endometrioid; Endometrial Neoplasms; Female; Humans; Prognosis; Retrospective Studies
PubMed: 35732320
DOI: 10.21873/cgp.20338