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Giordano G, Guareschi D, Thai E. Uterine Tumor Resembling Ovarian Sex-Cord Tumor (UTROSCT): A Rare Polyphenotypic Neoplasm. Diagnostics (Basel) 2024; 14:1271. [PMID: 38928686 PMCID: PMC11203005 DOI: 10.3390/diagnostics14121271] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/12/2024] [Revised: 05/24/2024] [Accepted: 05/31/2024] [Indexed: 06/28/2024] Open
Abstract
Uterine tumor resembling ovarian sex-cord tumor (UTROSCT) is a rare form of uterine mesenchymal neoplasm. Although UTROSCT generally exhibits benign behavior with a favorable prognosis, this neoplasm is nevertheless classified as being of uncertain malignant potential, given its low rate of recurrence and the fact that it rarely produces metastases (e.g., in the lymph nodes, epiploic appendix, omentum, small bowel, subcutaneous tissue, lungs). Its histogenesis is also uncertain. Typically, UTROSCT occurs in peri-menopausal or menopausal women, but it can sometimes be observed in young women. Usually, this neoplasm can be found in the uterine corpus as a nodular intramural lesion, while it is less frequently submucosal, subserosal, or polypoid/intracavitary. UTROSCT can cause abnormal bleeding, pelvic pain, enlarged uterus, and mass sensation, but sometimes it is found purely by chance. This neoplasm can be considered polyphenotypic on morphological, immunohistochemical, and genetic analyses. Generally, upon microscopic examination, UTROSCT shows a predominant pattern of the cords, nests, and trabeculae typical of sex-cord tumors of the ovary, while immunohistochemically it is characterized by a coexpression of epithelial, smooth muscle, and sex-cord markers. The aim of this review is to report clinical and pathological data and genetic alterations to establish their impact on the prognosis and management of patients affected by this rare entity.
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Affiliation(s)
- Giovanna Giordano
- Department of Medicine and Surgery, Pathology Unit, University of Parma, Viale A. Gramsci, 14, 43126 Parma, Italy; (D.G.); (E.T.)
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Shibahara M, Kurita T, Harada H, Tsuda Y, Hisaoka M, Yoshino K. Therapeutic management of uterine tumours resembling ovarian sex cord tumours including a focus on fertility: A systematic review. Eur J Obstet Gynecol Reprod Biol 2024; 295:1-7. [PMID: 38310674 DOI: 10.1016/j.ejogrb.2024.01.039] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/16/2023] [Revised: 12/14/2023] [Accepted: 01/29/2024] [Indexed: 02/06/2024]
Abstract
OBJECTIVE Uterine tumours resembling ovarian sex cord tumours (UTROSCTs) are extremely rare. To date, most patients with UTROSCTs have undergone hysterectomy and had a benign clinical course. Fertility-preserving surgery should be considered because some patients with UTROSCTs are aged < 40 years. This paper reviews the treatment and prognosis for patients with UTROSCTs, with a focus on fertility. METHODS PubMed, MEDLINE and Scopus were searched systematically for case reports and case series of UTROSCTs published in English from inception to December 2022, and initial treatment and recurrence rates were compared. The following data were extracted: age; symptoms; initial therapy; metastasis at diagnosis; disease-free survival (DFS); and recurrence. RESULTS In total, 147 patients (72 studies) reporting the clinical course of UTROSCTs were analysed. The median age at diagnosis was 50 years, and 28 (19.0 %) patients were aged < 40 years. Most patients (n = 125, 85.0 %) underwent hysterectomy as the initial surgery, with a recurrence rate of 17.6 % (n = 22). The recurrence rate was 30 % (n = 6) in patients who underwent mass resection (n = 20). Among the 15 patients who underwent mass resection aged < 40 years, seven went on to achieve pregnancy (46.7 %) and six had successful deliveries (40.0 %). No significant differences in 5- and 10-year DFS were found between the hysterectomy and mass resection groups (p = 0.123 and 0.0612, respectively). Bilateral salpingo-oophorectomy in addition to hysterectomy was not significantly associated with 10-year DFS (p = 0.548). CONCLUSION While total hysterectomy is the recommended treatment for UTROSCTs based on recurrence rates, mass resection is an acceptable treatment option for patients who wish to retain their childbearing potential. It is recommended that these women should plan for pregnancy and delivery as soon as possible after mass resection, and should undergo hysterectomy within 5 years.
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Affiliation(s)
- Mami Shibahara
- Department of Obstetrics and Gynaecology, University of Occupational and Environmental Health, Kitakyushu, Fukuoka, Japan
| | - Tomoko Kurita
- Department of Obstetrics and Gynaecology, University of Occupational and Environmental Health, Kitakyushu, Fukuoka, Japan.
| | - Hiroshi Harada
- Department of Obstetrics and Gynaecology, University of Occupational and Environmental Health, Kitakyushu, Fukuoka, Japan
| | - Yojiro Tsuda
- Department of Pathology and Oncology, University of Occupational and Environmental Health Japan, Kitakyushu, Fukuoka, Japan
| | - Masanori Hisaoka
- Department of Pathology and Oncology, University of Occupational and Environmental Health Japan, Kitakyushu, Fukuoka, Japan
| | - Kiyoshi Yoshino
- Department of Obstetrics and Gynaecology, University of Occupational and Environmental Health, Kitakyushu, Fukuoka, Japan
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Lin J, Liu L, Wang L, Ma N, Zhang K, Xie N, Yu H, Deng S, Sun Y. The management of uterine tumor resembling an ovarian sex cord tumor (UTROSCT): case series and literature review. World J Surg Oncol 2024; 22:42. [PMID: 38310233 PMCID: PMC10837875 DOI: 10.1186/s12957-024-03319-3] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/03/2023] [Accepted: 01/23/2024] [Indexed: 02/05/2024] Open
Abstract
AIMS To present a case series of 11 rare uterine tumors resembling ovarian sex cord tumors (UTROSCTs), and review the literature on this topic to offer up-to-date treatment management for UTROSCTs. METHOD Eight cases from Fujian Cancer Hospital between January 2017 and May 2023 and three patients from Fujian Union Hospital between October 2012 and October 2020 were retrospectively reviewed. All cases were pathologically confirmed as UTROSCTs by two senior and experienced pathologists. Clinical behaviors, medical data, histopathological features, therapy approaches, and survival outcomes were discussed. RESULTS The median age at initial diagnosis was 53 years (29-70 years). 3 (27.3%) patients were under 40. Seven cases presented with abnormal vaginal bleeding, one with menstrual disorder, one with abnormal vaginal secretion, and two patients were accidentally found by physical examination without any symptoms. Three patients were initially misdiagnosed with endometrial cancer by MRI. Curettage was performed in all cases. Nine of them were well diagnosed by routine curettage, except for two samples, which were identified after surgery. Immunohistochemical biomarkers, such as CD99, Desmin, WT-1, CK, Vimentin, SMA, α-Inhibin, Ki67, CD56, ER, PR, and CR, tend to be positive in UTRO SCs patients. Six patients underwent hysterectomy with bilateral salpingo-oophorectomy. Two cases received a radical hysterectomy with bilateral salpingo-oophorectomy, retroperitoneal lymph node dissection, and omentum dissection. Three UTROSCTs were under observation after mass resection. The median PFS was 24 months (range 1-125 months). CONCLUSION UTROSCT is a rare mesenchymal tumor with low malignant potential. Treatment modalities should be carefully considered to balance the therapy outcomes and patient needs. Surgery conservative management might be suitable for young women with fertility desires.
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Affiliation(s)
- Jie Lin
- Department of Gynecology, Clinical Oncology School of Fujian Medical University, Fujian Cancer Hospital, Fuzhou, Fujian Province, China
| | - Linying Liu
- Department of Gynecology, Clinical Oncology School of Fujian Medical University, Fujian Cancer Hospital, Fuzhou, Fujian Province, China
| | - Linghua Wang
- Department of Gynecology, Clinical Oncology School of Fujian Medical University, Fujian Cancer Hospital, Fuzhou, Fujian Province, China
| | - Ning Ma
- Department of Radiology, Clinical Oncology School of Fujian Medical University, Fujian Cancer Hospital, Fuzhou, Fujian Province, China
| | - Kailin Zhang
- Department of Pathology, Fujian Medical University Union Hospital, Fuzhou, China
| | - Ning Xie
- Department of Gynecology, Clinical Oncology School of Fujian Medical University, Fujian Cancer Hospital, Fuzhou, Fujian Province, China
| | - Haijuan Yu
- Department of Gynecology, Clinical Oncology School of Fujian Medical University, Fujian Cancer Hospital, Fuzhou, Fujian Province, China
| | - Sufang Deng
- Department of Gynecology, Clinical Oncology School of Fujian Medical University, Fujian Cancer Hospital, Fuzhou, Fujian Province, China
| | - Yang Sun
- Department of Gynecology, Clinical Oncology School of Fujian Medical University, Fujian Cancer Hospital, Fuzhou, Fujian Province, China.
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Watrowski R, Palumbo M, Guerra S, Gallo A, Zizolfi B, Giampaolino P, Bifulco G, Di Spiezio Sardo A, De Angelis MC. Uterine Tumors Resembling Ovarian Sex Cord Tumors (UTROSCTs): A Scoping Review of 511 Cases, Including 2 New Cases. MEDICINA (KAUNAS, LITHUANIA) 2024; 60:179. [PMID: 38276058 PMCID: PMC10820159 DOI: 10.3390/medicina60010179] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 12/02/2023] [Revised: 01/10/2024] [Accepted: 01/15/2024] [Indexed: 01/27/2024]
Abstract
Uterine Tumors Resembling Ovarian Sex Cord Tumors (UTROSCTs) are rare uterine mesenchymal neoplasms with uncertain biological potential. These tumors, which affect both premenopausal and postmenopausal women, usually have a benign clinical course. Nevertheless, local recurrences and distant metastases have been described. By analyzing 511 cases retrieved from individual reports and cases series, we provide here the most comprehensive overview of UTROSCT cases available in the literature, supplemented by two new cases of UTROSCTs. Case 1 was an asymptomatic 31-year-old woman who underwent a laparoscopic resection of a presumed leiomyoma. Case 2 was a 58-year-old postmenopausal woman with abnormal vaginal bleeding who underwent an outpatient hysteroscopic biopsy of a suspicious endometrial area. In both cases, immunohistochemical positivity for Calretinin and Inhibin was noted, typical for a sex cord differentiation. In both cases, total laparoscopic hysterectomy with bilateral salpingo-oophorectomy was performed. In light of the available literature, no pathognomonic clinical or imaging finding can be attributed to UTROSCT. Patients usually present with abnormal uterine bleeding or pelvic discomfort, but 20% of them are asymptomatic. In most cases, a simple hysterectomy appears to be the appropriate treatment, but for women who wish to become pregnant, uterus-preserving approaches should be discussed after excluding risk factors. Age, tumor size, lymphovascular space invasion, nuclear atypia, and cervical involvement are not reliable prognostic factors in UTROSCT. The current research suggests that aggressive cases (with extrauterine spread or recurrence) can be identified based on a distinct genetic and immunohistochemical phenotype. For instance, UTROSCTs characterized by GREB1::NCOA1-3 fusions and PD-L1 molecule expression appear to be predisposed to more aggressive behaviors and recurrence, with GREB1::NCOA2 being the most common gene fusion in recurrent tumors. Hence, redefining the criteria for UTROSCTs may allow a better selection of women suitable for fertility-sparing treatments or requiring more aggressive treatments in the future.
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Affiliation(s)
- Rafał Watrowski
- Department of Obstetrics and Gynecology, Helios Hospital Müllheim, 79379 Müllheim, Germany
- Faculty of Medicine, University of Freiburg, 79106 Freiburg, Germany
| | - Mario Palumbo
- Department of Public Health, School of Medicine, University of Naples Federico II, 80138 Naples, Italy; (M.P.); (S.G.); (A.G.); (B.Z.); (P.G.); (G.B.); (A.D.S.S.); (M.C.D.A.)
| | - Serena Guerra
- Department of Public Health, School of Medicine, University of Naples Federico II, 80138 Naples, Italy; (M.P.); (S.G.); (A.G.); (B.Z.); (P.G.); (G.B.); (A.D.S.S.); (M.C.D.A.)
| | - Alessandra Gallo
- Department of Public Health, School of Medicine, University of Naples Federico II, 80138 Naples, Italy; (M.P.); (S.G.); (A.G.); (B.Z.); (P.G.); (G.B.); (A.D.S.S.); (M.C.D.A.)
| | - Brunella Zizolfi
- Department of Public Health, School of Medicine, University of Naples Federico II, 80138 Naples, Italy; (M.P.); (S.G.); (A.G.); (B.Z.); (P.G.); (G.B.); (A.D.S.S.); (M.C.D.A.)
| | - Pierluigi Giampaolino
- Department of Public Health, School of Medicine, University of Naples Federico II, 80138 Naples, Italy; (M.P.); (S.G.); (A.G.); (B.Z.); (P.G.); (G.B.); (A.D.S.S.); (M.C.D.A.)
| | - Giuseppe Bifulco
- Department of Public Health, School of Medicine, University of Naples Federico II, 80138 Naples, Italy; (M.P.); (S.G.); (A.G.); (B.Z.); (P.G.); (G.B.); (A.D.S.S.); (M.C.D.A.)
| | - Attilio Di Spiezio Sardo
- Department of Public Health, School of Medicine, University of Naples Federico II, 80138 Naples, Italy; (M.P.); (S.G.); (A.G.); (B.Z.); (P.G.); (G.B.); (A.D.S.S.); (M.C.D.A.)
| | - Maria Chiara De Angelis
- Department of Public Health, School of Medicine, University of Naples Federico II, 80138 Naples, Italy; (M.P.); (S.G.); (A.G.); (B.Z.); (P.G.); (G.B.); (A.D.S.S.); (M.C.D.A.)
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Ferrara M, Pecorino B, D’Agate MG, Angelico G, Capoluongo ED, Malapelle U, Pepe F, Scollo P, Mereu L. Uterine Tumours Resembling Ovarian Sex-Cord Tumors: A Case Report and Review of the Literature. J Clin Med 2023; 12:7131. [PMID: 38002745 PMCID: PMC10671931 DOI: 10.3390/jcm12227131] [Citation(s) in RCA: 2] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/13/2023] [Revised: 10/18/2023] [Accepted: 11/14/2023] [Indexed: 11/26/2023] Open
Abstract
Uterine tumors resembling ovarian sex-cord tumors (UTROSCT) are thought to develop from pluripotent uterine mesenchymal cells or endometrial stromal cells with secondary sex-cord differentiation. The patient was a 73-year-old postmenopausal woman who had abnormal vaginal bleeding, and she underwent a laparoscopic hysterectomy with bilateral salpingo-oophorectomy. The diagnosis was a case of UTROSCT. A scoping review of the UTROSCT case report present in the literature has been conducted, and 63 articles were found, of which 45 were considered for the 66 clinical cases examined. At the time of diagnosis, six metastatic localizations were found in 59 patients undergoing demolitive surgery (10.2%). Recurrences were diagnosed in 13/59 (22%) patients with multiple locations. A molecular study was performed in 18/66 cases (27.3%) and genetic alterations were found in 10/18 (55.6%) patients. UTROSCTs are considered rare uterine tumors, typically with a favorable prognosis, and are generally considered to have a good prognosis. But, from the review done, they may already manifest themselves at advanced stages, with the possibility of recurrences even at a distance. It would, therefore, be important to be able to define the most aggressive forms and, perhaps, molecular investigation with sequencing could help identify patients most at risk.
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Affiliation(s)
- Martina Ferrara
- Department of Obstetrics and Gynecology, Cannizzaro Hospital, University of Enna “Kore”, 94100 Enna, Italy; (B.P.); (M.G.D.); (P.S.)
| | - Basilio Pecorino
- Department of Obstetrics and Gynecology, Cannizzaro Hospital, University of Enna “Kore”, 94100 Enna, Italy; (B.P.); (M.G.D.); (P.S.)
| | - Maria Gabriella D’Agate
- Department of Obstetrics and Gynecology, Cannizzaro Hospital, University of Enna “Kore”, 94100 Enna, Italy; (B.P.); (M.G.D.); (P.S.)
| | - Giuseppe Angelico
- Department of Anatomic Pathology and Histology, Cannizzaro Hospital, 95100 Catania, Italy;
| | | | - Umberto Malapelle
- Department of Public Health, University of Naples Federico II, 80126 Naples, Italy; (U.M.); (F.P.)
| | - Francesco Pepe
- Department of Public Health, University of Naples Federico II, 80126 Naples, Italy; (U.M.); (F.P.)
| | - Paolo Scollo
- Department of Obstetrics and Gynecology, Cannizzaro Hospital, University of Enna “Kore”, 94100 Enna, Italy; (B.P.); (M.G.D.); (P.S.)
| | - Liliana Mereu
- Department of Obstetrics and Gynecology, Policlinico G Rodolico, CHIRMED, University of Catania, 95124 Catania, Italy
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Abstract
RATIONALE A uterine tumor resembling an ovarian sex cord tumor (UTROSCT) is a clinically rare disease with an unclear origin and biological behavior. PATIENT CONCERNS We present a case of UTROSCT in a 42-year-old woman who presented with abnormally increased menstrual volume for 2 years. DIAGNOSES Initially, only ultrasound examination was performed to diagnose uterine fibroids, and then the tumor was surgically removed and sent for pathological examination. The patient was ultimately diagnosed with UTROSCT mainly based on pathological immunohistochemical examination and was further diagnosed with low malignant potential for recurrence based on genetic testing. INTERVENTIONS AND OUTCOMES The patient underwent hysterectomy and bilateral adnexectomy, and no adjuvant radiotherapy or chemotherapy was performed after the surgery. Follow-up to date has indicated that she is in good condition. LESSONS UTROSCT is a rare disease that requires pathological immunohistochemical examination to confirm the diagnosis and genetic testing when necessary so that a clear diagnosis can inform better decision-making regarding treatment measures.
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Affiliation(s)
- Xue-Ying Wang
- Department of Ultrasound, Affiliated Hospital of North Sichuan Medical College, Nanchong, Sichuan ProvinceChina
| | - Mao-Chun Zhang
- Department of Obstetrics and Gynecology, Affiliated Hospital of North Sichuan Medical College, Nanchong, Sichuan ProvinceChina
- *Correspondence: Maochun Zhang, Department of Obstetrics and Gynecology, Affiliated Hospital of North Sichuan Medical College, Nanchong, Sichuan Province 637000, China (e-mail: )
| | - Jiao Chen
- Department of Obstetrics and Gynecology, Affiliated Hospital of North Sichuan Medical College, Nanchong, Sichuan ProvinceChina
| | - Jiang-Hua Huang
- Department of Ultrasound, the Second Clinical Medical College of North Sichuan Medical College, Nanchong, Sichuan ProvinceChina
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Abstract
This review focuses on recent advances in epithelioid and myxoid uterine mesenchymal neoplasms, a category of tumors whereby diagnostic criteria have been rapidly evolving due to advances in molecular testing. Pertinent clinicopathological and molecular features are highlighted for perivascular epithelioid cell tumors, uterine tumors resembling ovarian sex cord tumors, BCOR/BCORL1-altered high-grade endometrial stromal sarcomas, and inflammatory myofibroblastic tumors. Novel developments in epithelioid and myxoid leiomyosarcomas are briefly discussed, and differential diagnoses with key diagnostic criteria are provided for morphologic mimickers.
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Affiliation(s)
- Elizabeth C Kertowidjojo
- Department of Pathology, University of Chicago Medicine, 5837 South Maryland Avenue, MC 6101, Chicago, IL 60637, USA
| | - Jennifer A Bennett
- Department of Pathology, University of Chicago Medicine, 5837 South Maryland Avenue, MC 6101, Chicago, IL 60637, USA.
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Zhou FF, He YT, Li Y, Zhang M, Chen FH. Uterine tumor resembling an ovarian sex cord tumor: A case report and review of literature. World J Clin Cases 2021; 9:6907-6915. [PMID: 34447841 PMCID: PMC8362530 DOI: 10.12998/wjcc.v9.i23.6907] [Citation(s) in RCA: 9] [Impact Index Per Article: 2.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/25/2021] [Revised: 06/02/2021] [Accepted: 06/17/2021] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Endometrial stromal tumors originate from the endometrial stroma and account for < 2% of all uterine tumors. Uterine tumor resembling an ovarian sex cord tumor (UTROSCT) is a rare histological class of endometrial stromal and related tumors according to the latest World Health Organization classification of female genital tumors. Here, we report a case of UTROSCT in a 51-year-old woman.
CASE SUMMARY A 51-year-old woman had irregular menses for 6 mo. The patient visited a local hospital for vaginal bleeding. Pelvic computed tomography (CT) showed a mass in the pelvic cavity. Five days later, she came to our hospital for further diagnosis. The results of contrast-enhanced CT and pelvic ultrasound at our hospital suggested a malignant pelvic tumor. She then underwent total removal of the uterus with bilateral salpingectomy. Postoperative histological examination showed that the tumor cells had abundant cytoplasm, ovoid and spindle-shaped nuclei, fine chromatin, a high nucleoplasm ratio, and a lamellar distribution. The findings were consistent with UTROSCT, and the results of immunohistochemical analysis supported that diagnosis. The tumor was International Federation of Gynecology and Obstetrics stage IB. No adjuvant therapy was administered after radical surgery. The patient was followed up for 58 mo, and no recurrence was found.
CONCLUSION We report a case of UTROSCT with abnormal menstruation as a symptom, which is one of the most common symptoms. In patients with vaginal bleeding, ultrasonography can be used as a screening test because of its convenience, speed, and lack of radiation exposure. For patients with long-term tamoxifen use, routine monitoring of the endometrium is recommended. As UTROSCT may have low malignant potential, surgery remains the primary management strategy. Additionally, fertility preservation in patients of childbearing age is a vital consideration.
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Affiliation(s)
- Fang-Fang Zhou
- Department of Ultrasound, Lishui Central Hospital, The Fifth Affiliated Hospital of Wenzhou Medical University, Lishui 323000, Zhejiang Province, China
| | - Ying-Tao He
- Department of Ultrasound, Lishui Central Hospital, The Fifth Affiliated Hospital of Wenzhou Medical University, Lishui 323000, Zhejiang Province, China
| | - Ying Li
- Department of Ultrasound, Lishui Central Hospital, The Fifth Affiliated Hospital of Wenzhou Medical University, Lishui 323000, Zhejiang Province, China
| | - Min Zhang
- Department of Pathology, Lishui Central Hospital, The Fifth Affiliated Hospital of Wenzhou Medical University, Lishui 323000, Zhejiang Province, China
| | - Fang-Hong Chen
- Department of Ultrasound, Lishui Central Hospital, The Fifth Affiliated Hospital of Wenzhou Medical University, Lishui 323000, Zhejiang Province, China
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Angelico G, Santoro A, Inzani F, Straccia P, Spadola S, Arciuolo D, Valente M, D’Alessandris N, Benvenuto R, Travaglino A, Raffone A, Zannoni GF. An Emerging Anti-p16 Antibody-BC42 Clone as an Alternative to the Current E6H4 for Use in the Female Genital Tract Pathological Diagnosis: Our Experience and a Review on p16ink4a Functional Significance, Role in Daily-Practice Diagnosis, Prognostic Potential, and Technical Pitfalls. Diagnostics (Basel) 2021; 11:diagnostics11040713. [PMID: 33923427 PMCID: PMC8073999 DOI: 10.3390/diagnostics11040713] [Citation(s) in RCA: 4] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/26/2021] [Revised: 04/09/2021] [Accepted: 04/14/2021] [Indexed: 01/10/2023] Open
Abstract
Background: To date, useful diagnostic applications of p16 IHC have been documented in gynecological pathology both for HPV-related and non-HPV-related lesions. In the present article, we reported our experience with the novel anti-p16 INK4a antibody (clone BC42), whose expression was tested across all different gynecologic neoplasms; we also compared it to the traditional E6H4 clone. Moreover, we discussed and explored all the diagnostic applications of p16 IHC in gynecologic pathology. Methods: Consultation cases covering a 5-year period (2016–2020) regarding gynecological neoplastic and non-neoplastic lesions in which immunohistochemistry for p16, clone E6H4 was originally performed, were retrospectively retrieved from the files of our institution. Immunohistochemical staining for p16ink4a (BC42) [Biocare Medical group-Paceco USA; Bioptica Milan] and p16ink4a (E6H4) [Ventana Medical Systems-Arizona USA; Roche] was performed by using the Ventana automated immunostainer (Ventana Medical Systems, Tucson, AZ, USA). The immunostaining pattern was defined as negative, focal/patchy, or diffuse. Results: A total of 196 cases, represented by 36 high-grade SIL/CIN3 of the uterine cervix, 30 cervical adenocarcinomas, 22 cervical squamous cell carcinoma, 70 endometrial carcinomas, 25 high grade serous ovarian carcinomas, 6 uterine adenomatoid tumors, and 10 uterine leiomyosarcomas were included in this study. Results showed concordant staining quality of both clones on all tested neoplastic tissues. Conclusions: The novel anti-p16 antibody (BC42 clone) appeared as an alternative to the current E6H4 for use in gynecological neoplasms, offering similar levels of positivity and equally reliable staining results.
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Affiliation(s)
- Giuseppe Angelico
- Unità di Gineco-Patologia e Patologia Mammaria, Dipartimento Scienze della Salute della Donna, del Bambino e di Sanità Pubblica, Fondazione Policlinico Universitario A. Gemelli IRCCS, 00168 Roma, Italy; (G.A.); (A.S.); (F.I.); (P.S.); (S.S.); (D.A.); (M.V.); (N.D.); (R.B.)
| | - Angela Santoro
- Unità di Gineco-Patologia e Patologia Mammaria, Dipartimento Scienze della Salute della Donna, del Bambino e di Sanità Pubblica, Fondazione Policlinico Universitario A. Gemelli IRCCS, 00168 Roma, Italy; (G.A.); (A.S.); (F.I.); (P.S.); (S.S.); (D.A.); (M.V.); (N.D.); (R.B.)
| | - Frediano Inzani
- Unità di Gineco-Patologia e Patologia Mammaria, Dipartimento Scienze della Salute della Donna, del Bambino e di Sanità Pubblica, Fondazione Policlinico Universitario A. Gemelli IRCCS, 00168 Roma, Italy; (G.A.); (A.S.); (F.I.); (P.S.); (S.S.); (D.A.); (M.V.); (N.D.); (R.B.)
| | - Patrizia Straccia
- Unità di Gineco-Patologia e Patologia Mammaria, Dipartimento Scienze della Salute della Donna, del Bambino e di Sanità Pubblica, Fondazione Policlinico Universitario A. Gemelli IRCCS, 00168 Roma, Italy; (G.A.); (A.S.); (F.I.); (P.S.); (S.S.); (D.A.); (M.V.); (N.D.); (R.B.)
| | - Saveria Spadola
- Unità di Gineco-Patologia e Patologia Mammaria, Dipartimento Scienze della Salute della Donna, del Bambino e di Sanità Pubblica, Fondazione Policlinico Universitario A. Gemelli IRCCS, 00168 Roma, Italy; (G.A.); (A.S.); (F.I.); (P.S.); (S.S.); (D.A.); (M.V.); (N.D.); (R.B.)
| | - Damiano Arciuolo
- Unità di Gineco-Patologia e Patologia Mammaria, Dipartimento Scienze della Salute della Donna, del Bambino e di Sanità Pubblica, Fondazione Policlinico Universitario A. Gemelli IRCCS, 00168 Roma, Italy; (G.A.); (A.S.); (F.I.); (P.S.); (S.S.); (D.A.); (M.V.); (N.D.); (R.B.)
| | - Michele Valente
- Unità di Gineco-Patologia e Patologia Mammaria, Dipartimento Scienze della Salute della Donna, del Bambino e di Sanità Pubblica, Fondazione Policlinico Universitario A. Gemelli IRCCS, 00168 Roma, Italy; (G.A.); (A.S.); (F.I.); (P.S.); (S.S.); (D.A.); (M.V.); (N.D.); (R.B.)
| | - Nicoletta D’Alessandris
- Unità di Gineco-Patologia e Patologia Mammaria, Dipartimento Scienze della Salute della Donna, del Bambino e di Sanità Pubblica, Fondazione Policlinico Universitario A. Gemelli IRCCS, 00168 Roma, Italy; (G.A.); (A.S.); (F.I.); (P.S.); (S.S.); (D.A.); (M.V.); (N.D.); (R.B.)
| | - Roberta Benvenuto
- Unità di Gineco-Patologia e Patologia Mammaria, Dipartimento Scienze della Salute della Donna, del Bambino e di Sanità Pubblica, Fondazione Policlinico Universitario A. Gemelli IRCCS, 00168 Roma, Italy; (G.A.); (A.S.); (F.I.); (P.S.); (S.S.); (D.A.); (M.V.); (N.D.); (R.B.)
| | - Antonio Travaglino
- Anatomic Pathology Unit, Department of Advanced Biomedical Sciences, University of Naples Federico II, 80125 Naples, Italy;
| | - Antonio Raffone
- Gynecology and Obstetrics Unit, Department of Neuroscience, Reproductive Sciences and Dentistry, University of Naples Federico II, 80125 Naples, Italy;
| | - Gian Franco Zannoni
- Unità di Gineco-Patologia e Patologia Mammaria, Dipartimento Scienze della Salute della Donna, del Bambino e di Sanità Pubblica, Fondazione Policlinico Universitario A. Gemelli IRCCS, 00168 Roma, Italy; (G.A.); (A.S.); (F.I.); (P.S.); (S.S.); (D.A.); (M.V.); (N.D.); (R.B.)
- Istituto di Anatomia Patologica, Università Cattolica del Sacro Cuore, 00168 Roma, Italy
- Correspondence: ; Tel.: +39-06-30154433
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Carbone MV, Cavaliere AF, Fedele C, Vidiri A, Aciuolo D, Zannoni G, Scambia G. Uterine tumor resembling ovarian sex-cord tumor: Conservative surgery with successful delivery and case series. Eur J Obstet Gynecol Reprod Biol 2020; 256:326-332. [PMID: 33264692 DOI: 10.1016/j.ejogrb.2020.11.043] [Citation(s) in RCA: 10] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/26/2020] [Revised: 10/18/2020] [Accepted: 11/13/2020] [Indexed: 12/15/2022]
Abstract
Uterine tumor resembling ovarian sex-cord tumor (UTROSCT) is a rare distinct cancer included in the current World Health Organization classification of endometrial stromal tumors. A battery of immunohistochemical markers are necessary for accurate diagnosis. Although few case reports and case series have been documented and therefore providing robust prognostic information several authors agree to consider UTROSCT as a low malignant potential tumour. In literature only five cases of conservative management were reported. We reported our experience and a review of conservative cases of literature. In this article we describe a series of 10 cases of UTROSCT and their clinical and pathologic feature. We report two cases of conservative surgical approach obtaining a successfully pregnancies. All of the patients are still alive. In conclusion, fertility sparing surgery should be offered to patients who wish preserve their fertility however radical surgery must be considerate after childbirth. Close follow-up is required for all patients due to the lack of prognostic biomarkers and long follow up is needed to evaluate safety of conservative surgery.
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Affiliation(s)
- Maria Vittoria Carbone
- Department of Woman and Child Health, Fondazione Policlinico Universitario A. Gemelli IRCCS, Italy.
| | - Anna Franca Cavaliere
- Department of Woman and Child Health, Fondazione Policlinico Universitario A. Gemelli IRCCS, Italy.
| | - Camilla Fedele
- Department of Woman and Child Health, Fondazione Policlinico Universitario A. Gemelli IRCCS, Italy.
| | - Annalisa Vidiri
- Department of Woman and Child Health, Fondazione Policlinico Universitario A. Gemelli IRCCS, Italy.
| | - Damiano Aciuolo
- Department of Woman and Child Health, Fondazione Policlinico Universitario A. Gemelli IRCCS, Italy.
| | - Gianfranco Zannoni
- Department of Woman and Child Health, Fondazione Policlinico Universitario A. Gemelli IRCCS, Italy; Università Cattolica del Sacro Cuore, Rome, Italy.
| | - Giovanni Scambia
- Department of Woman and Child Health, Fondazione Policlinico Universitario A. Gemelli IRCCS, Italy; Università Cattolica del Sacro Cuore, Rome, Italy.
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11
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Chang B, Bai Q, Liang L, Ge H, Yao Q. Recurrent uterine tumors resembling ovarian sex-cord tumors with the growth regulation by estrogen in breast cancer 1-nuclear receptor coactivator 2 fusion gene: a case report and literature review. Diagn Pathol 2020; 15:110. [PMID: 32921307 PMCID: PMC7489201 DOI: 10.1186/s13000-020-01025-8] [Citation(s) in RCA: 24] [Impact Index Per Article: 4.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/07/2020] [Accepted: 09/03/2020] [Indexed: 12/16/2022] Open
Abstract
BACKGROUND Uterine tumors resembling ovarian sex-cord tumors (UTROSCTs) are rare mesenchymal neoplasms predominantly arising in perimenopausal and postmenopausal women. UTROSCTs with growth regulation by estrogen in breast cancer 1 (GREB1)-rearrangement or GREB1-rearranged uterine tumors are exceptionally rare, with only 12 previously reported cases. Here, we report a case of UTROSCT with the GREB1-nuclear receptor coactivator 2 (NCOA2) fusion gene. CASE PRESENTATION A 57-year-old woman presented with a 10.0 cm uterine mass. The tumor was composed of short spindle or epithelioid cells, arranged in diffused sheets, nested, and trabecular/cordlike. The tumor harbored the GREB1-NCOA2 fusion gene, as confirmed by RNA sequencing. The tumor recurred in the pelvis at 30 months after the initial diagnosis. We also compared the clinical and pathologic features of this case with those of the 12 previously published uterine GREB1-rearranged tumors. Of the combined 13 cases (present case and 12 previous cases), the mean age of patients was 64.8 years (range, 51-74 years). Of the nine reported cases of GREB1-rearranged tumor with follow up, four cases recurred or metastasized (44.4%). Microscopically, most tumors (10/12, 83.3%) showed infiltrative growth, and two were well demarcated. Mitotic figures ranged from 0 to 14 per 10 high-power fields (2 mm2; mean: 3.6). Lymphovascular invasion and necrosis were each present in two cases (2/12, 16.7% and 2/7, 28.6%, respectively). CONCLUSIONS This case provided further evidence that UTROSCTs with GREB1-rearrangement may have a high risk of recurrence/metastasis. Further studies are necessary to clarify the clinical features of this type of tumor, particularly the prognosis, potential treatment, and range of possible molecular events.
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Affiliation(s)
- Bin Chang
- Department of Pathology, Fudan University Shanghai Cancer Center, Shanghai, China. .,Department of Oncology, Shanghai Medical College, Fudan University, Shanghai, China.
| | - Qianming Bai
- Department of Pathology, Fudan University Shanghai Cancer Center, Shanghai, China.,Department of Oncology, Shanghai Medical College, Fudan University, Shanghai, China
| | - Lin Liang
- Department of Pathology, Fudan University Shanghai Cancer Center, Shanghai, China.,Department of Oncology, Shanghai Medical College, Fudan University, Shanghai, China
| | - Huijuan Ge
- Department of Pathology, Fudan University Shanghai Cancer Center, Shanghai, China.,Department of Oncology, Shanghai Medical College, Fudan University, Shanghai, China
| | - Qianlan Yao
- Department of Pathology, Fudan University Shanghai Cancer Center, Shanghai, China.,Department of Oncology, Shanghai Medical College, Fudan University, Shanghai, China
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