Abstract
Background
Müllerian adenosarcoma is a biphasic neoplasm most commonly occurring in the uterus
and less frequently of the ovary. It has been rarely described to occur in other sites
such as peritoneum and liver.
Patients and Methods
In this study, we report the clinicopathologic, immunohistochemical and molecular
features of a primary Müllerian adenosarcoma of the urinary bladder in a 62-year-old
woman. To our knowledge, this is the first report of detailed pathologic characterization
of Müllerian adenosarcoma primary to the urinary bladder in the literature.
Results
Light microscopy showed a biphasic epithelial and stromal tumor with benign-appearing
glands surrounded by densely cellular endometrial-type stroma that is densely cellular
with increased mitotic figures. The stroma surrounding the glands was more cellular
than the intervening areas, which were more loose and edematous. Immunohistochemistry
staining revealed positive staining for Pax-2/8 within the glands, for positive CD10
and WT-1 within the spindle cell stroma, and for estrogen and progesterone receptors
in both. Staining for desmin, GATA3, p63, and human papillomavirus was negative. Molecular
analyses identified mutations in protein kinase B E17K, fms related tyrosine kinase
3 D835N, KRAS proto-oncogene, GTPase G12D, and HRAS proto-oncogene, GTPase G12S. These
novel molecular aberrations have yet to be reported in the medical literature. X chromosome
inactivation analysis revealed a clonal pattern in the stromal component and a nonclonal
pattern in the epithelial component. Currently, the patient is disease/recurrence-free
after regular follow-up of approximately 84 months.
Conclusion
This case represents, to our knowledge, the first reported diagnosis of Müllerian
adenosarcoma arising in the urinary bladder with extensive clinicopathologic, immunohistochemical,
and molecular analyses.
Keywords
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References
- Müllerian adenosarcoma of the uterus. A clinicopathologic analysis of ten cases of a distinctive type of Müllerian mixed tumor.Cancer. 1974; 34: 1138-1149
- Müllerian adenosarcoma of the uterus: a clinicopathologic analysis of 100 cases with a review of the literature.Hum Pathol. 1990; 21: 363-381
- Müllerian adenosarcomas of the uterus with sarcomatous overgrowth. A clinicopathological analysis of 10 cases.Am J Surg Pathol. 1989; 13: 28-38
- Mesodermal (Müllerian) adenosarcoma of the ovary: a clinicopathologic analysis of 40 cases and a review of the literature.Am J Surg Pathol. 2002; 26: 1243-1258
- Extrauterine mesodermal (Müllerian) adenosarcoma: a clinicopathologic analysis of five cases.Am J Clin Pathol. 1978; 69: 276-283
- Extragenital Müllerian adenosarcoma with pouch of Douglas location.BMC Cancer. 2011; 11: 171
- Extragenital adenosarcoma arising in the pouch of Douglas.Gynecol Oncol. 1994; 53: 373-375
- Vaginal adenosarcoma arising from endometriosis.Gynecol Oncol. 2000; 76: 123-125
- Adenosarcoma arising in hepatic endometriosis.Eur Radiol. 2000; 10: 1287-1289
- Müllerian adenosarcoma of the uterus with sarcomatous overgrowth following tamoxifen treatment for breast cancer.Rev Hosp Clin Fac Med Sao Paulo. 2000; 55: 17-20
- Müllerian adenosarcoma of the uterine corpus associated with tamoxifen therapy: a report of six cases and a review of tamoxifen-associated endometrial lesions.Int J Gynecol Pathol. 1996; 15: 222-229
- Müllerian adenosarcoma of the uterus associated with tamoxifen therapy.Arch Gynecol Obstet. 2000; 264: 105-107
- Uterine Müllerian adenosarcoma following adenomyoma in a woman on tamoxifen therapy.Gynecol Oncol. 1992; 44: 104-109
- Endometrial adenosarcoma with adjuvant tamoxifen therapy for primary breast carcinoma.Australas Radiol. 1998; 42: 157-158
- Müllerian adenosarcoma of the uterus in association with tamoxifen therapy.Histopathology. 2000; 36: 91-92
- Copy number estimation algorithms and fluorescence in situ hybridization to describe copy number alterations in human tumors.Pathol Int. 2009; 59: 218-228
- Virtual-karyotyping with SNP microarrays in morphologically challenging renal cell neoplasms: a practical and useful diagnostic modality.Am J Surg Pathol. 2009; 33: 1276-1286
- Optimization of the Affymetrix GeneChip Mapping 10K 2.0 Assay for routine clinical use on formalin-fixed paraffin-embedded tissues.Diagn Mol Pathol. 2008; 17: 3-13
- Genetically heterogeneous and clonally unrelated metastases may arise in patients with cutaneous melanoma.Am J Surg Pathol. 2007; 31: 1029-1037
- Evidence for common clonal origin of multifocal lung cancers.J Natl Cancer Inst. 2009; 101: 560-570
- Histogenesis of sarcomatoid urothelial carcinoma of the urinary bladder: evidence for a common clonal origin with divergent differentiation.J Pathol. 2007; 211: 420-430
- Mesodermal adenosarcoma of the testis.Am J Surg Pathol. 2005; 29: 420-423
- Recurrent Müllerian adenosarcoma like tumor of seminal vesicle.Indian J Pathol Microbiol. 2010; 53: 342-344
- Müllerian adenosarcomalike tumor of the seminal vesicle. A case report with immunohistochemical and ultrastructural observations.Arch Pathol Lab Med. 1992; 116: 1072-1076
- Cystic epithelial-stromal tumor of the seminal vesicle.Am J Surg Pathol. 1987; 11: 210-217
- WHO classification of female reproductive organs.IARC, Lyon, France2014
- Endometrioid adenosarcoma of the bladder arising from endometriosis.J Urol. 1990; 143: 813-815
- Laparoscopic management of 15 patients with infiltrating endometriosis of the bladder and a case of primary intravesical endometrioid adenosarcoma.Fertil Steril. 2002; 78: 872-875
- Involvement of chromosome 8 in müllerianadenosarcoma.Int J Gynecol Pathol. 2017; 36: 24-30
- Genomewide copy number analysis of Müllerian adenosarcoma identified chromosomal instability in the aggressive subgroup.Mod Pathol. 2016; 29: 1070-1082
- Targeted genomic analysis of Müllerian adenosarcoma.J Pathol. 2015; 235: 37-49
- Uterine adenosarcomas are mesenchymal neoplasms.J Pathol. 2016; 238: 381-388
- Adenosarcoma of the uterus: a clinicopathologic, DNA flow cytometric, p53 and mdm-2 analysis of 11 cases.Int J Gynecol Cancer. 1999; 9: 37-43
- Exome sequencing identifies frequent mutation of the SWI/SNF complex gene PBRM1 in renal carcinoma.Nature. 2011; 469: 539-542
- Smooth muscle neoplasms of the urinary bladder: a clinicopathologic study of 51 cases.Am J Surg Pathol. 2010; 34: 502-509
- Sarcomatoid carcinoma of the urinary bladder: the final common pathway of urothelial carcinoma dedifferentiation.Am J Surg Pathol. 2011; 35: e34-e46
- Tumor-like lesions of the urinary bladder.Mod Pathol. 2009; 22: S37-S52
- Clear cell adenocarcinoma of the bladder and urethra. A report of three cases and review of the literature.Am J Surg Pathol. 1985; 9: 816-826
- Endometrial stromal sarcoma involving the urinary bladder: a study of 6 cases.Am J Surg Pathol. 2014; 38: 982-989
- Polypoid endometriosis: a clinicopathologic analysis of 24 cases and a review of the literature.Am J Surg Pathol. 2004; 28: 285-297
- Long-term outcome and natural history of uterine adenosarcomas.Gynecol Oncol. 2010; 119: 305-308
- Bladder endometriosis: a systematic review of pathogenesis, diagnosis, treatment, impact on fertility, and risk of malignant transformation.Eur Urol. 2017; 71: 790-807
- Ureteral endometriosis: clinicopathological and immunohistochemical study of 7 cases.Hum Pathol. 2008; 39: 954-959
Article info
Publication history
Published online: May 24, 2017
Accepted:
May 16,
2017
Received:
May 7,
2017
Identification
Copyright
© 2017 Elsevier Inc. All rights reserved.