Pregnancy with recurrent endometrial stromal sarcoma: a case report / 中华围产医学杂志

This paper reported the clinical characteristics, diagnosis, and treatment of a case of recurrent endometrial stromal sarcoma with term pregnancy. The patient had undergone laparoscopic surgery to remove hysteromyoma before conception in 2017, which was pathologically diagnosed as low-grade endometrial stromal sarcoma after surgery. Due to her strong reproductive willingness, the patient attempted to conceive in light of her stable condition and no evidence of recurrence and was closely followed up with an informed choice. She conceived successfully in 2020 and underwent regular pregnancy examinations. Ultrasound examination at 37 +4 weeks of gestation revealed a slightly hypoechoic mass of about 6.3 cm×4.5 cm size in the pelvic cavity. After admission, a pelvic MRI indicated multiple solid nodules in the right adnexa uteri and beside the iliac vessels in the left pelvic wall and anterior pelvic wall with the larger one being about 58 mm×28 mm. Diffusion-weighted imaging showed multiple pelvic nodules and masses with significant diffusion restriction. The patient was diagnosed as having multiple solid nodules and masses in the pelvic cavity, and the recurrence of sarcoma was highly suspected. Brain CT and lung CT showed no obvious metastatic lesions. A consultation involving the Department of Gynecological Tumor Chemoradiotherapy was held and the sarcoma recurrence during pregnancy was prenatally diagnosed. After ruling out the contraindications for surgery, a cesarean section was performed in the lower segment of uterus under general anesthesia and a live female baby was delivered at 38 ＋3 weeks. The excised mass was confirmed as recurrent uterine stromal sarcoma by rapid freezing pathology during cesarean section. A combination surgery was performed subsequently, including total extra-fascial hysterectomy, bilateral oophorectomy, bilateral salpingectomy, appendectomy, greater omentum resection, pelvic lesion resection (right side), and pelvic adhesiolysis. Recurrent low-grade uterine stromal sarcoma was reconfirmed by postoperative pathology. The patient was discharged after recovery. After two years of follow-up, no distant metastasis recurrences were found.

Nódulo del estroma endometrial: Reporte de un caso infrecuente / Endometrial stromal nodule: A rare case report

Introducción: Los tumores del estroma endometrial representan menos del 2% de los tumores uterinos, estando dentro de las neoplasias menos comunes del cuerpo uterino. Se pueden dividir en cuatro categorías principales: nódulo del estroma endometrial, sarcoma del estroma endometrial de bajo grado, sarcoma del estroma endometrial de alto grado y sarcoma uterino indiferenciado. En el presente trabajo se describe el caso de un paciente con diagnóstico de nódulo del estroma endometrial. Caso clínico : Paciente femenino de 50 años de edad, quien refiere inicio de enfermedad en marzo de 2022, caracterizado por presentar sangrado uterino anormal anemizante y aumento de volumen abdominal, por lo que acude a facultativo foráneo, donde indican paraclínicos. Para el día 12 de julio de 2022, presentó dolor abdominal de aparición brusca de moderada a severa intensidad. Motivo por el cual acudió a nuestro centro. Se determina anemia y leucocitosis. Estudios de imagen reportan masa voluminosa, densidad mixta, bien delimitada. Otra lesión hiperecogénica, que corresponde a quiste unicameral de ovario derecho. Se decide resolución quirúrgica, mediante la realización de laparotomía exploradora más protocolo de endometrio, con evolución satisfactoria de la paciente. Conclusión : La histerectomía es el tratamiento de elección. El estudio anatomopatológico es fundamental para su diagnóstico final y diferenciación de los sarcomas estromales, ya que su pronóstico, tratamiento y seguimiento es diferente(AU)

Introduction: Endometrial stromal tumors represent less than 2% of uterine tumors, being among the least common neoplasms of the uterine body. They can be divided into four main categories: endometrial stromal nodule, low-grade endometrial stromal sarcoma, high-grade endometrial stromal sarcoma and undifferentiated uterine sarcoma. This paper describes the case of a patient with a diagnosis of endometrial stromal nodule.Clinical case : A 50-year-old female patient, who reported the onset of the disease in March 2022, characterized by abnormal uterine bleeding with anemia and increased abdominal volume, for which she went to a foreign physician, where they indicated paraclinical tests. On July 12, 2022, he presented abdominal pain of sudden onset of moderate to severe intensity. Which is why she came to our center. Anemia and leukocytosis are determined. Imaging studies report a voluminous mass, mixed density, well delimited. Another hyperechoic lesion, which corresponds to a unicameral cyst of the right ovary. Surgical resolution was decided by performing an exploratory laparotomy and endometrial protocol, with satisfactory evolution of the patient.Conclusion : Hysterectomy is the treatment of choice. The anatomopathological study is fundamental for its final diagnosis and differentiation of stromal sarcomas, since its prognosis, treatment and follow-up are different(AU)

Two Cases of Uterine Tumors Resembling Ovarian Sex-cord Tumors: Rare Case of Uterine Tumor / 이화의대지

Uterine tumors resembling ovarian sex-cord tumors (UTROSCT) are very rare tumors that occur mainly in the uterine fundus of women in reproductive age. These tumors can be classified into group 1 and group 2 by histological results. In group 1, epithelial-like differentiation is partially observed in the tumors. In group 2, sex-cord elements are predominant in uterine mural mass. We experienced UTROSCT group 1 in a 29-year-old woman who complained of severe abdominal pain that started one week after delivery and UTROSCT group 2 case in a 49-year-old woman who complained of dysfunctional uterine bleeding. We report two different types of UTROSCT cases that we experienced.

Ultrasonographic findings of low-grade endometrial stromal sarcoma of the uterus with a focus on cystic degeneration

PURPOSE: The goal of this study was to perform a retrospective analysis of the ultrasonographic findings associated with low-grade endometrial stromal sarcoma. METHODS: Ten pathologically confirmed cases of low-grade endometrial stromal sarcoma at our institution from January 2007 to April 2014 were retrospectively reviewed. All patients underwent a preoperative transvaginal ultrasound. Two radiologists came to a consensus regarding the location, size, margin, and echogenicity of the tumor, as well as the presence of intratumoral cystic degeneration and its extent and configuration. RESULTS: Low-grade endometrial stromal sarcoma manifested as an intramural mass protruding into the endometrial cavity (n=6) or as a purely intramural mass (n=4). The maximal diameter of the lesion ranged from 4 to 9.1 cm (mean, 6.2 cm). The imaging features of low-grade endometrial stromal sarcoma were variable: six cases involved predominantly solid masses containing cystic degeneration, one was a predominantly unilocular cystic mass, two were ill-defined infiltrative solid masses, and one was a well-defined solid mass. Among the seven cases with internal cystic degeneration, five patients showed a multiseptated cystic area or a cystic area with multiple small clusters, while a unilocular cystic area within the tumor was found in two patients. CONCLUSION: Low-grade endometrial stromal sarcoma is associated with variable ultrasonographic findings with regard to the location, margin, and configuration of the lesion. Multiseptated cystic areas and multiple small areas of cystic degeneration are common.

A Case of Metastatic Low-Grade Endometrial Stromal Sarcoma Treated with Letrozole after Ovarian Ablation by Radiotherapy / Journal of the Korean Cancer Association, 대한암학회지

A 50-year-old woman was admitted to our hospital due to multiple lung nodules detected incidentally on a chest X-ray. A video-assisted thoracoscopic lung biopsy revealed low-grade endometrial stromal sarcoma (LG-ESS). She had undergone a simple hysterectomy 1 year earlier owing to a diagnosis of adenomyosis. A review of her previous hysterectomy specimen showed not endometriosis but LG-ESS. According to the patient's levels of serum follicle stimulating hormone and estradiol, she was in the premenopausal state with retained and normally functioning ovaries. She then underwent ovarian ablation by radiotherapy, after which she was administered 2.5 mg of letrozole once per day. Three months later, the size of the metastatic nodules in both lungs had decreased. The patient was followed up for 24 months while continuing on letrozole, and maintained a partial remission. We report herein on a case of metastatic LG-ESS treated with letrozole after ovarian ablation by radiotherapy.

Cyclin D1 and p16INK4 positive endometrial stromal sarcoma: A case report with new insights.

Endometrial stromal sarcoma (ESS) has a wide histopathological spectrum with CD10 as its diagnostic marker. Recently, few non-conventional ESSs have been identifi ed that lack diffuse CD10 expression. A 46-year-old, perimenopausal lady referred to us with history of vaginal bleeding. On clinical examination and radiological imaging, a polypoid endometrial tumor was identifi ed. Hysterectomy revealed a multinodular tumor in the myometrium. Microscopically, the tumor composed of rather banal oval to spindle-shaped cells in a fi bromyxoid stroma. Focal areas displayed compact cellular arrangement, unassociated with signifi cant mitoses and necrosis. Immunohistochemically, tumor cells were focally positive for CD10, estrogen receptor, progesterone receptor, p16INK4 and were diffusely positive for cyclinD1. Diagnosis of cyclinD1 and p16INK4 positive ESS was offered. This case highlights the value of additional IHC markers, especially cyclinD1 and p16INK4 in order to identify certain ESSs that lack diffuse CD10 immunoexpression; are invariably misdiagnosed as undifferentiated sarcomas, but actually form a relatively more aggressive subset of ESSs.

Sarcoma del estroma endometrial de grado alto / High degree endometrial stromal sarcoma

El sarcoma del estroma endometrial es un tumor infrecuente, que comprende menos del 1 % de los tumores malignos ginecológicos. Presentamos el caso de una adolescente de 16 años con hemorragia uterina anormal a la que se le realizó histerectomía subtotal. Microscópicamente, la neoplasia estaba constituida por una proliferación maligna de células estromales del endometrio. El estudio inmuno-histoquímico mostró positividad para vimentina, desmina, CD10, CD117. El CD10 es un marcador inmuno-histoquímico del estroma endometrial normal y de los tumores del estroma endometrial, y es útil para el diagnóstico diferencial entre el sarcoma del estroma endometrial y el leiomioma celular o el leiomiosarcoma uterino. Las mejores opciones de tratamiento se obtienen con un enfoque multidisciplinario y en centros especializados.

Endometrial stromal sarcoma is a rare tumor comprising less than 1 % of gynecological malignancies. We report the case of a 16 year-old adolescent with abnormal uterine bleeding who underwent hysterectomy subtotal. At microscopy the neoplasm was composed of malignant proliferation of endometrial stromal cells. Immunohistochemical studies were positive for vimentin, desmin, CD10 and CD117. CD10 is an immunohistochemical marker of normal endometrial stroma and of endometrial stromal neoplasms. This marker is useful in the differential diagnosis of endometrial stromal sarcoma versus uterine cellular leiomyoma or uterine leiomyosarcoma. The best treatment options are obtained with a focus multidisciplinary, in specialized centers.

Tumor misto de células musculares lisas e do estroma endometrial uterino: relato de caso / Mixed endometrial stromal and smooth muscle tumor of the uterus: case report

Os tumores mistos de células musculares lisas e do estroma endometrial uterino, caracterizados pela presença de componentes de ambas as linhagens, coexistindo em proporções quase equivalentes, são neoplasmas raros. Possuem potencial biológico incerto e se comportam de acordo com o componente estromal. A imuno-histoquímica é uma grande aliada no diagnóstico microscópico, pois a clínica e os exames de imagem não ajudam a diferenciá-los de outras doenças uterinas. Descrevemos o caso de uma paciente cuja hipótese diagnóstica era de leiomioma uterino e que, após cirurgia, foi diagnosticada pelo estudo anatomopatológico como tumor misto de células musculares lisas e do estroma endometrial uterino.

Mixed tumors of uterine smooth muscle and endometrial stromal cells, which are characterized by the presence of components from both cell lineages with similar proportions, are rare neoplasms. Their biological potential is uncertain, and they behave according to the stromal component. Immunohistochemistry is an important ally in microscopic diagnosis, because symptoms and imaging exams do not help in the differentiation from other uterine diseases. We describe a case of a patient who had been previously diagnosed with uterine leiomyoma, and after surgery, the anatomopathological study revealed a mixed tumor of uterine smooth muscle and endometrial stromal cells.

Efeito do anti-TNF-α em implantes endometriais no peritônio de ratas / Effect of anti-TNF-α on peritoneal endometrial implants of rats

OBJETIVO: Avaliar o efeito da terapia anti-TNF-α no tratamento de implantes endometriais no peritônio de ratas. MÉTODOS: Os implantes endometrióticos foram induzidos cirurgicamente em 120 ratas Wistar-Albino. Os animais foram aleatoriamente distribuídos em 4 grupos. O grupo C (n=36) recebeu uma injeção intraperitoneal de 0,2ml de solução salina. O grupo L (n=41) recebeu uma injeção subcutânea de 1mg/kg de leuprolide. O grupo I5 (n=20) recebeu uma injeção subcutânea de 5mg/kg de anticorpo monoclonal anti-fator de necrose tumoral (TNF) a (infliximab). O grupo I10 (n=20) recebeu uma injeção subcutânea de 10mg/kg de infliximab. As ratas foram sacrificadas após 21 dias para se avaliar o tamanho dos implantes e a expressão do TNF-α. RESULTADOS: O tratamento com leuprolide promoveu uma redução absoluta na área de superfície do implante comparado com o grupo C (+14mm vs. 0mm; p=0,013) e com o grupo I10 (+14mm vs. +5mm; p=0,018). Da mesma forma, uma redução percentual da area de superfície do implante foi observada comparando o grupo L com o grupo C (+33,3 por cento vs. 0 por cento; p=0,005) e com o grupo I10 (+33,3 por cento vs. +18,3 por cento; p=0,027). O tratamento com infliximab não foi capaz de diminuir a área de superfície do implante comparado com o grupo C. A expressão de TNF-α reduziu nos grupos L, I5 e I10 comparado com o grupo C (505,6µm² vs. 660,5µm² vs. 317,2µm² vs. 2519,3µm², respectivamente; p<0,001). CONCLUSÃO: A terapia anti-TNF-α reduziu a expressão de TNF-α nos implantes endometrióticos mas não reduziu a área de superfície da lesão.

OBJECTIVE: To evaluate the effect of anti-TNF-α in the treatment of endometrial implants in the peritoneum of rats. METHODS: Endometrial implants were surgically induced in 120 female Wistar-Albino rats. The animals were randomly divided into four groups. Group C (n = 36) received an intraperitoneal injection of 0.2 ml of saline. Group L (n = 41) received a subcutaneous injection of 1mg/kg of leuprolide. Group I5 (n = 20) received a subcutaneous injection of 5mg/kg of monoclonal anti-tumor necrosis factor (TNF) a (infliximab). Group I10 (n = 20) received a subcutaneous injection of 10mg/kg of infliximab. The rats were sacrificed after 21 days to assess the size of the implants and the expression of TNF. RESULTS: Treatment with leuprolide (group L) promoted an absolute reduction in the surface area of the implant when compared with group C (+14 mm vs. 0mm, p = 0.013) and group I10 (+14 mm vs. +5 Mm, p = 0.018). Likewise, a percentage reduction of surface area of the implant was observed comparing group L with group C (+33.3 percent vs. 0 percent, p = 0.005) and group I10 (+33.3 percent vs. +18.3 percent, p = 0.027). Treatment with infliximab was not able to decrease the surface area of the implants when compared with group C. The expression of TNF-α in groups L, I5 and I10 was lower than in group C (505.6 mm² vs. 660.5 mm² vs. 317.2 mm² vs. 2519.3 mm², respectively; p <0.001). CONCLUSION: The anti-TNF-α therapy reduced the expression of TNF-α in endometriotic implants, but did not reduce the surface area of the lesion.