Small cell carcinoma of the ovary, hypercalcemic type: a mini review

Small cell carcinoma of the ovary, hypercalcemic type (SCCOHT) is a rare and highly aggressive ovarian neoplasm, predominantly affecting young women, often in their second or third decade of life. Despite its distinctive clinical and pathological features, diagnosis is frequently delayed due to over...

Full description

Saved in:
Bibliographic Details
Main Authors: Marta Tripepi, Ana G. da Costa, Dennis S. Chi, Jorge Lima, João Casanova
Format: Article
Language:English
Published: Frontiers Media S.A. 2025-08-01
Series:Frontiers in Oncology
Subjects:
Online Access:https://www.frontiersin.org/articles/10.3389/fonc.2025.1645361/full
Tags: Add Tag
No Tags, Be the first to tag this record!
_version_ 1849405226612686848
author Marta Tripepi
Ana G. da Costa
Dennis S. Chi
Dennis S. Chi
Jorge Lima
Jorge Lima
João Casanova
author_facet Marta Tripepi
Ana G. da Costa
Dennis S. Chi
Dennis S. Chi
Jorge Lima
Jorge Lima
João Casanova
author_sort Marta Tripepi
collection DOAJ
description Small cell carcinoma of the ovary, hypercalcemic type (SCCOHT) is a rare and highly aggressive ovarian neoplasm, predominantly affecting young women, often in their second or third decade of life. Despite its distinctive clinical and pathological features, diagnosis is frequently delayed due to overlapping characteristics with other small round blue cell tumors. A hallmark of SCCOHT is the biallelic inactivation of the SMARCA4 gene, which leads to loss of BRG1 protein expression and disrupts epigenetic regulation via the SWI/SNF chromatin-remodeling complex. Unlike many other malignancies, SCCOHT exhibits low mutational burden and diploid karyotype, suggesting that epigenetic dysregulation, rather than genomic instability, is the underlying oncogenic mechanism. Clinically, SCCOHT often presents with nonspecific abdominal or pelvic symptoms and is uniquely associated with paraneoplastic hypercalcemia in up to two-thirds of cases. Diagnosis requires a combination of imaging, laboratory evaluation, histopathology, and immunohistochemistry. Treatment is not standardized but typically involves a multimodal approach, including radical surgery and platinum-based chemotherapy, often with multi-agent regimens. The role of radiotherapy is less well defined but may be considered for local control or palliation. Prognosis remains poor, with high recurrence rates and limited response to salvage therapy. Emerging molecular insights have prompted investigations into targeted therapies and immunotherapy, though clinical data are limited. Given the frequent presence of germline SMARCA4 mutations, genetic counseling is strongly recommended, and ongoing research is essential to improve diagnostic accuracy, personalize treatment, and enhance outcomes for this devastating malignancy.
format Article
id doaj-art-e7c3647ebae64c7389d247d231539c19
institution Kabale University
issn 2234-943X
language English
publishDate 2025-08-01
publisher Frontiers Media S.A.
record_format Article
series Frontiers in Oncology
spelling doaj-art-e7c3647ebae64c7389d247d231539c192025-08-20T03:36:44ZengFrontiers Media S.A.Frontiers in Oncology2234-943X2025-08-011510.3389/fonc.2025.16453611645361Small cell carcinoma of the ovary, hypercalcemic type: a mini reviewMarta Tripepi0Ana G. da Costa1Dennis S. Chi2Dennis S. Chi3Jorge Lima4Jorge Lima5João Casanova6Department of Women and Children’s Health, Clinic of Gynecology and Obstetrics, University of Padua, Padua, ItalyGynecologic Oncology Unit, Obstetrics and Gynecology Service, Department of Surgery, Hospital da Luz Lisboa, Lisbon, PortugalGynecology Service, Department of Surgery, Memorial Sloan Kettering Cancer Center, New York, NY, United StatesDepartment of OB/GYN, Weill Cornell Medical College, New York, NY, United StatesObstetrics and Gynecology Service, Department of Surgery, Hospital da Luz Lisboa, Lisbon, PortugalComprehensive Health Research Centre (CHRC), NOVA Medical School, Faculdade de Ciências Médicas, NOVA Medical School (NMS), Faculdade de Ciencias Medicas (FCM), Universidade Nova De Lisboa, Lisbon, PortugalGynecologic Oncology Unit, Obstetrics and Gynecology Service, Department of Surgery, Hospital da Luz Lisboa, Lisbon, PortugalSmall cell carcinoma of the ovary, hypercalcemic type (SCCOHT) is a rare and highly aggressive ovarian neoplasm, predominantly affecting young women, often in their second or third decade of life. Despite its distinctive clinical and pathological features, diagnosis is frequently delayed due to overlapping characteristics with other small round blue cell tumors. A hallmark of SCCOHT is the biallelic inactivation of the SMARCA4 gene, which leads to loss of BRG1 protein expression and disrupts epigenetic regulation via the SWI/SNF chromatin-remodeling complex. Unlike many other malignancies, SCCOHT exhibits low mutational burden and diploid karyotype, suggesting that epigenetic dysregulation, rather than genomic instability, is the underlying oncogenic mechanism. Clinically, SCCOHT often presents with nonspecific abdominal or pelvic symptoms and is uniquely associated with paraneoplastic hypercalcemia in up to two-thirds of cases. Diagnosis requires a combination of imaging, laboratory evaluation, histopathology, and immunohistochemistry. Treatment is not standardized but typically involves a multimodal approach, including radical surgery and platinum-based chemotherapy, often with multi-agent regimens. The role of radiotherapy is less well defined but may be considered for local control or palliation. Prognosis remains poor, with high recurrence rates and limited response to salvage therapy. Emerging molecular insights have prompted investigations into targeted therapies and immunotherapy, though clinical data are limited. Given the frequent presence of germline SMARCA4 mutations, genetic counseling is strongly recommended, and ongoing research is essential to improve diagnostic accuracy, personalize treatment, and enhance outcomes for this devastating malignancy.https://www.frontiersin.org/articles/10.3389/fonc.2025.1645361/fullsmall cell ovarian cancerhypercalcemic typehypercalcemiaSMARCA4ovarian cancerpregnancy
spellingShingle Marta Tripepi
Ana G. da Costa
Dennis S. Chi
Dennis S. Chi
Jorge Lima
Jorge Lima
João Casanova
Small cell carcinoma of the ovary, hypercalcemic type: a mini review
Frontiers in Oncology
small cell ovarian cancer
hypercalcemic type
hypercalcemia
SMARCA4
ovarian cancer
pregnancy
title Small cell carcinoma of the ovary, hypercalcemic type: a mini review
title_full Small cell carcinoma of the ovary, hypercalcemic type: a mini review
title_fullStr Small cell carcinoma of the ovary, hypercalcemic type: a mini review
title_full_unstemmed Small cell carcinoma of the ovary, hypercalcemic type: a mini review
title_short Small cell carcinoma of the ovary, hypercalcemic type: a mini review
title_sort small cell carcinoma of the ovary hypercalcemic type a mini review
topic small cell ovarian cancer
hypercalcemic type
hypercalcemia
SMARCA4
ovarian cancer
pregnancy
url https://www.frontiersin.org/articles/10.3389/fonc.2025.1645361/full
work_keys_str_mv AT martatripepi smallcellcarcinomaoftheovaryhypercalcemictypeaminireview
AT anagdacosta smallcellcarcinomaoftheovaryhypercalcemictypeaminireview
AT dennisschi smallcellcarcinomaoftheovaryhypercalcemictypeaminireview
AT dennisschi smallcellcarcinomaoftheovaryhypercalcemictypeaminireview
AT jorgelima smallcellcarcinomaoftheovaryhypercalcemictypeaminireview
AT jorgelima smallcellcarcinomaoftheovaryhypercalcemictypeaminireview
AT joaocasanova smallcellcarcinomaoftheovaryhypercalcemictypeaminireview