Low-grade endometrial stromal sarcoma with extensive sex cord differentiation, heterologous elements, and complex atypical hyperplasia: Case report and review of literature

Richmond, Abby M.; Rohrer, Andrew J.; Davidson, Susan A.; Post, Miriam D.

doi:10.1016/j.gore.2016.12.002

Cited by 15 publications

(11 citation statements)

References 8 publications

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“…Immunohistochemically, the sex cord elements in LGESS show positive staining of endometrial stromal, smooth muscle markers. Sex cord markers including inhibin, CD99, calretinin and CD56 could also be positive [19,20]. PHF1 rearrangement has been found to be predominant in the sex cord variant of…”

Section: Discussionmentioning

confidence: 98%

Corded and hyalinized endometrioid carcinoma: a rare case and review of the literature

Lang

Yuan

et al. 2020

Preprint

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Background Corded and hyalinized endometrioid carcinoma(CHEC) is a rare morphological variation of endometrioid carcinoma(EC) in the endometrium. Reports of CHEC were very limited. We represent the clinical and pathological findings of this rare endometrioid carcinoma in a 26-year-old woman and reviews the literatures updated on CHEC. Case presentation: A 26-year-old woman presented with abnormal vaginal bleeding for 3 months and the initial cervical biopsy revealed a mullerian mixed tumor in another clinic. Abdominopelvic computed tomography revealed a mass in the uterine cavity and cervix, suggesting a malignant tumor. Histologically, the tumor showed a biphasic pattern characterized by an appearance of 2 components, the conventional endometrioid carcinoma component and sex cord-like component with hyalinization. In the areas of sex cord-like elements, the epithelioid and spindle cells were usually seen around the glands, and mostly arranged in cords or trabeculaes, sometimes embedded within a richly hyalinized collagenous or sometimes myxoid matrix. Tumor cells in the sex cord-like region show a different immunohistochemical expression pattern from conventional adenocarcinoma. Cytokeratin(CK) and vimentin are positive in both components, and vimentin shows more diffuse positivity while CK is more restricted and focally expressed in tumor cells in the sex cord-like region. Complete loss of expression of E-cadherin and epithelial membrane antigen(EMA) was seen in tumor cells in the sex cord-like region whereas it was well preserved in the area of conventional adenocarcinoma. Nuclear expression of β-catenin was noted in tumor cells in the sex cord-like region. P53 was focally positive in both components. Based on histological and immunochemical examinations, the patient was diagnosed with CHEC. Conclusions CHEC is not uncommonly mistaken for a wide variety of diseases. It’s of great significance to raise the awareness of CHEC to avoid over-treatment caused by over-diagnosis, especially in young patients and in curettage. We report one case of CHEC in a 26-year-old woman which was misdiagnosed as a mullerian mixed tumor in the initial curettage specimen. The clinicopathologic, light microscopic, immunohistochemical features of this tumor are described and the differential diagnosis is discussed.

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Section: Discussionmentioning

confidence: 98%

Corded and hyalinized endometrioid carcinoma: a rare case and review of the literature

Lang

Yuan

et al. 2020

Preprint

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“…Although some tumor cells were morphologically characterized as having cord-like differentiation in trabecular, cord, nested, or small tubular structural arrangements, no Leydig cell or Sertoli cell-like tubular sex cord-like structures were observed. Studying cases with LG-ESS-associated sex cord-like differentiation revealed that the available literature included mainly case reports, with no systematic research[3-7]. Regarding how to assess the sex cord-like differentiation in LG-ESS, we believe that in addition to the presence of the sex cord-like differentiated structures, including the trabecular, cord, or small tubular structures in the LG-ESS background, immunohistochemical assays are conducive to making assessments.…”

Section: Discussionmentioning

confidence: 99%

Metastatic low-grade endometrial stromal sarcoma with sex cord and smooth muscle differentiation: A case report

Zhu¹,

Sun²,

Xia³

et al. 2019

WJCC

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BACKGROUND Metastatic low-grade endometrial stromal sarcoma (LG-ESS) with sex cord-like and smooth muscle-like differentiation is rare. This article reports such a case with multiple recurrences and with extensive pelvic and abdominal metastasis. CASE SUMMARY A 47-year-old female patient was diagnosed with multiple cystic masses in the pelvic cavity by magnetic resonance imaging examination. Based on the postoperative pathological and immunohistochemical analyses of the surgical specimen, she was diagnosed with a metastatic low-grade endometrial stromal sarcoma with sex cord and smooth muscle differentiation. CONCLUSION LG-ESS is a low-grade malignant tumor with a high recurrence rate and metastasis probability. It is easily misdiagnosed initially. It is essential to distinguish LG-ESS with sex cord-like differentiation from uterine tumour resembling ovarian sex cord tumour.

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“…ESN and LGESS could also show sex cord-like features [38–40]. According to Clement and Scully, this variant usually comprises sex cord-like elements below 50% [1], however, LGESS with a sex cord-like contribution more than 50% has also been reported [41, 42]. The tumor cells in the sex cord-like elements are small and uniform and have round or oval nuclei with inconspicuous nucleoli and scanty cytoplasm (Fig.…”

Section: Introductionmentioning

confidence: 99%

“…Additionally, nuclear atypia and mitotic activity are not prominent [41, 43]. Moreover, large foam-like or Sertoli-like cells with abundant cytoplasm have also been reported [1, 42, 43]. Immunohistochemically, the sex cord elements show positive staining of epithelial, endometrial stromal, smooth muscle markers and hormone receptors, while epithelial membrane antigen (EMA) is always negative.…”

Section: Introductionmentioning

confidence: 99%

Uterine lesions with sex cord-like architectures: a systematic review

2019

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BackgroundSex cord-like elements are rarely observed in uterine lesions, but these morphological patterns could appear in a variety of uterine tumors and non-tumorous lesions. In this review, we collected the literatures regarding the uterine tumorous and non-tumorous lesions containing sex cord-like elements and summarized these lesions in terms of clinicopathological, immunohistochemical, and molecular features in order to further understand these lesions and provide some new ideas for differential diagnosis.Main bodyThis section provides a comprehensive overview of the clinicopathological, immunohistochemical, and molecular features of uterine lesions with sex cord-like architectures including uterine tumors resembling ovarian sex cord tumors, endometrial stromal tumors, adenomyosis, endometrial polyps, leiomyoma, epithelioid leiomyosarcoma, adenosarcoma, sertoliform endometrioid carcinoma, corded and hyalinized endometrioid carcinoma, mesonephric adenocarcinoma, and mesonephric-like adenocarcinoma. The differential diagnosis based on morphology, immunohistochemistry, and molecular alterations has also been discussed.ConclusionThe sex cord-like areas in these lesions show heterogeneous but similar morphological features. Additionally, immunohistochemical staining plays a limited role in differential diagnosis. Furthermore, it is of significance for pathologists to better understand these lesions in order to avoid confusion and mistakes during pathological diagnosis, especially in a biopsy/curettage specimen.

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Low-grade endometrial stromal sarcoma with extensive sex cord differentiation, heterologous elements, and complex atypical hyperplasia: Case report and review of literature

Cited by 15 publications

References 8 publications

Corded and hyalinized endometrioid carcinoma: a rare case and review of the literature

Corded and hyalinized endometrioid carcinoma: a rare case and review of the literature

Metastatic low-grade endometrial stromal sarcoma with sex cord and smooth muscle differentiation: A case report

Uterine lesions with sex cord-like architectures: a systematic review

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