Alveolar Soft Part Sarcoma of the Uterus: Case Report and Review of the Literature

Burch, David; Hitchcock, Andrew; Masson, G. M.

doi:10.1006/gyno.1994.1172

Cited by 16 publications

(5 citation statements)

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“…ASPS involvement of the female genital tract is uncommon, and only 30 cases have been reported so far, including our case (4–9). Of these, nine cases occurred in the uterine corpus, including five in the myometrium and four in the endometrium (Table 1).…”

Section: Discussionmentioning

confidence: 82%

“…Alveolar soft part sarcoma (ASPS) is a rare malignant soft tissue tumor, mainly localized in the extremities, occurring principally in adolescents and young adults (1–3). ASPS in the uterine corpus is quite rare; only eight such cases have been reported in the literature (4–9). Because of its rarity, it may be misdiagnosed, most often as epithelioid smooth‐muscle tumor, paraganglioma, metastatic renal cell carcinoma, or clear cell sarcoma.…”

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confidence: 99%

“…We here report another case of ASPS of the uterine corpus with typical histological features of ASPS and nuclear immunoreactivity for TFE3, which was recently described as a specific marker for ASPS (10). The histogenesis of uterine ASPS is still uncertain (4–9), though a myogenic origin has been most suggested (2, 3). In the female genital tract, it is well known that estrogen receptor (ER) and progesterone receptor (PgR) are expressed in most mesenchymal tumors and CD10 in endometrial stromal tumors (11).…”

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confidence: 99%

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Alveolar soft part sarcoma of the endometrium with expression of CD10 and hormone receptors

Kasashima

Minato

Kobayashi

et al. 2007

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Alveolar soft part sarcoma (ASPS) is a rare tumor of uncertain histogenesis, mainly localized in the extremities. ASPS originating in the uterine corpus is quite rare; only eight such cases have been reported in the literature. We here present another case of ASPS found in the endometrium in a 50-year-old woman. Metastatic malignant tumor, including ASPS from other organs, was excluded by physical examination and imaging modalities. Thallium 201 was only localized in the uterus. The tumor showed characteristic histological features of ASPS: alveolar architecture with fibrovascular septa and abundant eosinophilic granular cytoplasm with periodic acid-Schiff-positive crystalline material. Diffuse nuclear immunoreactivity for TFE3, a marker recently reported to be specific for ASPS, further supported the diagnosis of ASPS. Interestingly, this tumor was negative for myogenic markers, but positive for CD10, progesterone receptor, and estrogen receptor. These immunohistochemical results and the tumor location suggest a possible link between endometrial stromal cells and the development of this tumor.

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Section: Discussionmentioning

confidence: 82%

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confidence: 99%

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confidence: 99%

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Alveolar soft part sarcoma of the endometrium with expression of CD10 and hormone receptors

Kasashima

Minato

Kobayashi

et al. 2007

APMIS

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“…115 Approximately two-thirds of all reported gynecologic tract cases were primary to the uterus, 14 of which originated in the uterine cervix 6,116-118 and 9 of which originated in the uterine corpus. [119][120][121][122][123][124] The latter 9 cases are summarized in Table 4. The patients ranged in age from 14 to 50 years (mean 38 y).…”

Section: Pleomorphic Rhabdomyosarcomamentioning

confidence: 99%

Heterologous and Rare Homologous Sarcomas of the Uterine Corpus

Fadare¹

2011

Advances in Anatomic Pathology

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Pure sarcomas of the uterine corpus are uncommon, constituting less than 3% of all malignancies at this site, and most of them are leiomyosarcomas and endometrial stromal sarcomas. Rare histotypes of homologous sarcomas and heterologous sarcomas are occasionally encountered, and the absence of significant accumulated experience with these histotypes at this location may potentially raise diagnostic and patient management difficulties. In this article, the clinicopathologic attributes of all earlier reported sarcomas of the uterine corpus other than leiomyosarcomas and endometrial stromal sarcomas are summarized. Included are embryonal rhabdomyosarcoma, pleomorphic rhabdomyosarcoma, angiosarcoma, alveolar soft part sarcoma, malignant perivascular epithelioid cell tumors (PEComas), osteosarcoma, chondrosarcoma, liposarcomatous tumors, malignant extrarenal rhabdoid tumors, Ewing sarcoma/primitive neuroectodermal tumor, and other rare histotypes. Embryonal rhabdomyosarcoma (20%), Ewing sarcoma/primitive neuroectodermal tumor (17%), angiosarcoma (14%), and pleomorphic rhabdomyosarcoma (13%) appeared to be more common than the others, although there was no single overwhelmingly prevalent histotype in the group. A subset, including embryonal rhabdomyosarcoma, alveolar soft part sarcoma, and PEComas, peak in the premenopausal years, but most of the others were observed in postmenopausal women. Favorable outcomes have been reported for the patients diagnosed with alveolar soft part sarcoma, and the prognosis for their counterparts with PEComa remains a matter of debate. Multimodal therapeutic approaches to contemporary patients with embryonal rhabdomyosarcomas have resulted in significantly improved outcomes. Unfortunately, most of the other sarcomas have been associated with rapid tumor progression and unfavorable patient outcomes. The differential diagnosis for these sarcomas is often extensive and varies by histotype, but their accurate diagnosis fundamentally requires the careful exclusion of biphasic malignancies.

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“… PASDR crystals ND Intracytoplasmic crystalline inclusions Guillou et al (1991) [ 4 ] 3 × 2.5 × 2.5 cm well-circumscribed intramyometrial nodule Uncapsulated lesion pushing border delineated by endometrium, large cells with organoid arrangement, separated by fibrovascular septae, granular cytoplasms with crystals. Areas with nuclear pleomorphism POS Vim, focal POS HMB45, NKI/C3, patchy dot-like cytokeratin lw POS ND Burch et al (1994) [ 29 ] 3-cm endometrial polyp Large cells with granular cytoplasm, crystals on PAS D ND Intracytoplasmic crystalline inclusions Nielsen et al (1995) [ 30 ] 1-cm intramural nodule Large cell, arranged in organoid appearance separated by delicate fibrovascular stroma crystals on PAS D ND ND 3.5-cm submucosal nodule Large cell, arranged in organoid appearance separated by delicate fibrovascular stroma, crystals on PAS D ND ND Radig K et al (1998) [ 31 ] 3-cm well-circumscribed yellow-whitish, intramural nodule Pseudoalveolar and trabecular pattern, polygonal and round cells vesicular nuclei, with nucleoli, crystals on PAS D NEG for S100, Ck, EMA, POS caldes Intracytoplasmic crystalline inclusions 4.5-cm well-circumscribed, intramural nodule Pseudoalveolar pattern, granular ND Intracytoplasmic crystalline inclusions absent Kasashima S et al (2007) [ 5 ] 1.9 × 1.9 × 1.0 cm greyish, endometrial exophytic nodule Large cells with organoid arrangement, separated by fibrovascular septae, granular cytoplasms with crystals Nuclear POS: TFE3, PGR, ER, CD10 ND Vesicular nuclei and pleomorphic nuclei and multinucleated cells. Mitoses very rare.…”

Section: Case Presentationmentioning

confidence: 99%

Primary alveolar soft part sarcoma of uterine corpus: a case report with immunohistochemical, ultrastructural study and review of literature

et al. 2015

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BackgroundAlveolar soft part sarcoma (ASPS) is a rare mesenchymal malignancy. ASPS usually occurs most commonly in the deep soft tissues of the thigh and buttock or the head and neck regions.ASPS that originate from the uterine corpus are even more rare, with only 10 previous cases reported in the English literature.Case presentationIn our case, the alveolar features were completely lost and the tumour shows a solid, non-alveolar pattern and the nuclei have marked variation in nuclear size, and multinucleation. The correct pathological diagnosis has been made by immuno- histochemical and ultrastructural features, which rvealed overexpression of TFE3 and peculiar cytoplasmic crystalline inclusions.In this paper, an additional case of primary ASPS of uterine corpus is reported with immunohistochemical, ultrastructural study and review of literature in the effort to delineate its clinical and pathological features. In this unusual site, the diagnosis can be problematic because ASPS can mimic other primary or metastatic uterine neoplasms.ConclusionsThus, in this unusual presentation an essential diagnostic marker is the nuclear over-expression of TFE3 as well as ultrastructural study, which reveals the presence of peculiar cytoplasmic crystalline inclusions.

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Alveolar Soft Part Sarcoma of the Uterus: Case Report and Review of the Literature

Cited by 16 publications

References 0 publications

Alveolar soft part sarcoma of the endometrium with expression of CD10 and hormone receptors

Alveolar soft part sarcoma of the endometrium with expression of CD10 and hormone receptors

Heterologous and Rare Homologous Sarcomas of the Uterine Corpus

Primary alveolar soft part sarcoma of uterine corpus: a case report with immunohistochemical, ultrastructural study and review of literature

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