Uterine Epithelioid Trophoblastic Tumor in a 44-Year-Old Woman: A Diagnostic Dilemma

Background: Epithelioid trophoblastic tumor (ETT) is a rare and newly defined disease, which most commonly occurs in women of reproductive age and can be a sequela of any gestational event. ETT can be present in both intrauterine and extrauterine sites. Case report: A woman of reproductive age, without specific comorbidities and with a single pregnancy and natural childbirth eight years ago, was diagnosed initially with poorly differentiated pleomorphic leiomyosarcoma on the hemostatic uterine curettage. Conclusion: Our case highlights that ETT presents a diagnostic challenge due to its rarity and histologic resemblance to other pathologies. Misdiagnosis delays effective treatment and affects survival. To date, only 8 cases of ETT of the uterus without previous gestational event and normal human chorionic gonadotropin (β-HCG) levels in a 60-year literature survey have been reported.

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Epithelioid Trophoblastic Tumor: A Case Report and Review of the Literature

Case Reports in Obstetrics and Gynecology ◽

10.1155/2012/862472 ◽

2012 ◽

Vol 2012 ◽

pp. 1-5 ◽

Cited By ~ 2

Author(s):

Eirwen M. Scott ◽

Ashlee L. Smith ◽

Mohamed Mokhtar Desouki ◽

Alexander B. Olawaiye

Keyword(s):

Reproductive Age ◽

Women Of Reproductive Age ◽

Diagnostic Challenge ◽

Postoperative Evaluation ◽

Trophoblastic Tumor ◽

Laparoscopic Assisted Vaginal Hysterectomy ◽

Epithelioid Trophoblastic Tumor ◽

Abnormal Vaginal Bleeding ◽

Laparoscopic Assisted ◽

Disease Free

Epithelioid trophoblastic tumor (ETT) is a rare gestational trophoblastic tumor. Cases of ETT present as abnormal vaginal bleeding in women of reproductive age, with low human chorionic gonadotropin (hCG) levels. ETT can be a sequela of any gestational event and can present in both intrauterine and extrauterine sites. Metastasis and death have been reported. We present a case of a 44-year-old female incidentally diagnosed with ETT following laparoscopic-assisted vaginal hysterectomy. Postoperative evaluation for metastatic disease was negative. The patient has been closely followed and remains disease free 8 months postoperatively. ETT presents a diagnostic challenge due to its rarity and histologic resemblance to other pathologies. ETT is relatively chemoresistant and managed surgically. Misdiagnosis delays effective treatment and affects survival.

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Epithelioid Trophoblastic Tumor: Review of a Rare Neoplasm of the Chorionic-Type Intermediate Trophoblast

Archives of Pathology & Laboratory Medicine ◽

10.5858/2006-130-1875-ettroa ◽

2006 ◽

Vol 130 (12) ◽

pp. 1875-1877

Author(s):

Kimberly H. Allison ◽

Jason E. Love ◽

Rochelle L. Garcia

Keyword(s):

Gestational Trophoblastic Disease ◽

Reproductive Age ◽

Placental Lactogen ◽

Variable Expression ◽

Trophoblastic Tumor ◽

Trophoblastic Cells ◽

Epithelioid Trophoblastic Tumor ◽

Positive Immunostaining ◽

Nodular Growth ◽

Type Intermediate

Abstract We present a brief review of epithelioid trophoblastic tumor, a rare trophoblastic neoplasm derived from chorionic-type intermediate trophoblastic cells that typically presents in reproductive-age women between 1 and 18 years following a previous gestation. Histologic features include a nodular growth pattern of monomorphic, epithelioid cells within a hyaline matrix. Areas of necrosis and mitotic activity (0–9 mitoses per 10 high-power fields) are additional features of this neoplasm. Positive immunostaining for p63 and cytokeratin, frequent location in the lower uterine segment and endocervix, as well as the epithelioid appearance can lead to confusion with squamous cell carcinoma. Inhibin-α is typically expressed, as well as focal, more variable expression of other trophoblastic markers including β-human chorionic gonadotropin, human placental lactogen, placental alkaline phosphate, and Mel-CAM (CD148). The clinical behavior of this rare form of gestational trophoblastic disease is difficult to predict. Although most cases follow a benign course following resection, there is a potential for metastatic disease.

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Extrauterine Epithelioid Trophoblastic Tumor (ETT) Arising from an Antecedent Undiagnosed Molar Pregnancy Presenting as an Adnexal mass: A Case Report

American Journal of Clinical Pathology ◽

10.1093/ajcp/aqab191.164 ◽

2021 ◽

Vol 156 (Supplement_1) ◽

pp. S78-S78

Author(s):

J Gallardo ◽

K Hummel ◽

K McCluskey ◽

R Masand ◽

J Sunde

Keyword(s):

Case Report ◽

Tumor Cells ◽

Dna Fingerprinting ◽

Adnexal Mass ◽

Molar Pregnancy ◽

Fingerprinting Analysis ◽

Trophoblastic Tumor ◽

Eosinophilic Cytoplasm ◽

Epithelioid Trophoblastic Tumor ◽

Β Hcg

Abstract Introduction/Objective Epithelioid trophoblastic tumor (ETT) is an extremely rare neoplasm derived from chorionic type intermediate trophoblast. ETT usually follows an antecedent term pregnancy but can also follow spontaneous abortions or molar pregnancy. ETT most often arises from the endometrium, followed by the cervix. Extrauterine ETT are extremely rare, with few cases reported in literature. Methods/Case Report A 41-year-old woman with three term pregnancies presented with abdominal pain, ten years after her last pregnancy. Imaging findings of a 3.5 cm adnexal mass coupled with an elevated serum β-hCG (~ 900 mIU/ml), led to the suspicion of an ectopic pregnancy. Hysterectomy with salpingectomy revealed a 4.7 cm, tan- yellow, necrotic mass in the adnexal region abutting but distinct from the uterine serosa. Histologic evaluation showed a well- circumscribed tumor with pushing borders. The tumor cells were epithelioid with well-defined eosinophilic cytoplasm, monomorphic nuclei, frequent mitosis, and abundant geographic necrosis. The tumor cells were positive for β-hCG, GATA-3, PLAP and inhibin, with focal weak staining squamous markers p63 and p40. DNA fingerprinting analysis, performed to confirm the diagnosis of ETT, revealed a homozygous tumor with two copies of non-maternal genes indicating that the antecedent index gestation giving rise to the tumor was an undiagnosed hydatidiform mole. Following surgery, serum β-hCG levels were normal and the patient is currently on surveillance. Results (if a Case Study enter NA) NA Conclusion We present an extremely rare case of extrauterine ETT arising from a previously undetected molar pregnancy. The diagnosis should be suspected when a mass is observed at extrauterine sites with elevated β-hCG levels in patients with or without vaginal bleeding. Histologic differential of squamous cell carcinoma needs to be ruled out with immunostains. Due to its rarity and highly variable presentation, this entity remains a diagnostic challenge. DNA fingerprinting analysis demonstrating non-maternal genes can help confirm the diagnosis of ETT.

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Intestinal endometriosis: a diagnostic dilemma

International Journal of Reproduction Contraception Obstetrics and Gynecology ◽

10.18203/2320-1770.ijrcog20190894 ◽

2019 ◽

Vol 8 (3) ◽

pp. 1139

Author(s):

Sunil Kumar Juneja ◽

Satpal Singh Virk ◽

Saurabh Singla ◽

Kamal Sachdeva ◽

Harpreet Kaur ◽

...

Keyword(s):

Bowel Obstruction ◽

Gastrointestinal Symptoms ◽

Integrated Approach ◽

Reproductive Age ◽

Diagnostic Challenge ◽

Diagnostic Dilemma ◽

Intestinal Endometriosis ◽

True Incidence ◽

Intestinal Involvement ◽

Menstruating Women

Background: Endometriosis is a benign gynaecological condition that causes significant morbidity to women of reproductive age group. It uncommonly affects the gastrointestinal tract and acute bowel obstruction is a rare manifestation.Methods: A retrospective observational study was conducted on eight patients.Results: In three patients (37.5%), small intestinal involvement was seen, and colon was involved in five patients (62.5%). One (12.5%) patient presented with complete bowel obstruction while in others partial obstruction of the intestine was seen.Conclusions: Intestinal endometriosis is a diagnostic challenge and should be considered in young menstruating women with gastrointestinal symptoms. Intestinal endometriosis is the most common extra-pelvic site and it is found in 12% of women with endometriosis. The true incidence of endometriosis causing bowel obstruction is unknown. Pre or intraoperative sigmoidoscopy may prove helpful in ruling out malignancy. The gold standard for diagnosis is laparoscopy and biopsy, which allows a full assessment of the pelvis as well as surgical resection if required. The management of endometriosis is an integrated approach of both medical and surgical therapy. Bowel resection is usually undertaken if there are features of obstruction or bleeding, and if there is suspicion of malignancy.

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Epithelioid Trophoblastic Tumor: An Outcome-Based Literature Review of 78 Reported Cases

International Journal of Gynecological Cancer ◽

10.1097/igc.0b013e31829ea023 ◽

2013 ◽

Vol 23 (7) ◽

pp. 1334-1338 ◽

Cited By ~ 26

Author(s):

Xiaofei Zhang ◽

Weiguo Lü ◽

Bingjian Lü

Keyword(s):

Squamous Cell Carcinoma ◽

Cell Carcinoma ◽

Squamous Cell ◽

Figo Stage ◽

Lower Uterine Segment ◽

Reproductive Age ◽

Small Data ◽

Data Set ◽

Trophoblastic Tumor ◽

Epithelioid Trophoblastic Tumor

ObjectivesEpithelioid trophoblastic tumor (ETT) is very rare; and therefore, a substantially increased data set is unlikely to be obtained in the near future. This analysis aimed to assess the effects of current management on clinical outcomes and to identify potential prognostic indicators in ETT.MethodsWe applied a literature search using PubMed to analyze the clinical data of 78 published cases of ETT.ResultsWomen with ETT present at reproductive age (mean ± SD, 37.1 ± 8.7 years) and have a slightly to moderately elevated serum β-human chorionic gonadotropin (median, 665 IU/L). Epithelioid trophoblastic tumor is frequently present in the lower uterine segment/cervix (26/58 cases) and can be misdiagnosed as squamous cell carcinoma (6/26). Lung is the most common extrauterine site of ETT (5/11 with uterine ETT and 10/20 without uterine ETT). Kaplan-Meier analysis indicates that chemotherapy (surgery with postoperative chemotherapy vs surgery alone) is associated with increased ETT relapse (P= 0.005), even after stratification by International Federation of Gynecology and Obstetrics (FIGO) stage (P= 0.008); but FIGO stage remains the only significant prognostic indicator for ETT (P= 0.015).ConclusionsThis analysis confirms the hypothetical chemotherapy resistance and prognostic value of FIGO staging in ETT. These findings remain tentative given the small data set available for analysis and the reporting bias from these published cases; however, they may confer a risk-adapted therapy. Finally, both gynecologists and pathologists should be alert to the potential misdiagnosis of squamous cell carcinoma when ETT is present in the lower uterine segment/cervix.

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Vaginal carcinoma after hysterectomy for invasive hydatidiform mole: a diagnostic dilemma between epithelioid trophoblastic tumor and vaginal cancer

Pathology ◽

10.1097/01.pat.0000454414.23779.8f ◽

2014 ◽

Vol 46 ◽

pp. S91-S92

Author(s):

Nopporn Satabongkoch ◽

Sunida Rewsuwan ◽

Kornkanok Sukpan ◽

Kittipak Charoenkwan ◽

Surapan Khunamornpong

Keyword(s):

Hydatidiform Mole ◽

Vaginal Cancer ◽

Diagnostic Dilemma ◽

Vaginal Carcinoma ◽

Trophoblastic Tumor ◽

Epithelioid Trophoblastic Tumor

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Diagnosing and treating rare lesions in a low resource setting: lessons from a hybrid epithelioid trophoblastic tumor and choriocarcinoma

Ghana Medical Journal ◽

10.4314/gmj.v51i4.9 ◽

2018 ◽

Vol 51 (4) ◽

pp. 196-199

Author(s):

Patrick K Akakpo ◽

Kofi Ulzen-Appiah ◽

Evans Agbeno ◽

Leonard Derkyi-Kwarteng

Keyword(s):

Squamous Cell Carcinoma ◽

Cell Carcinoma ◽

Squamous Cell ◽

Histopathological Diagnosis ◽

Low Resource ◽

Trophoblastic Tumor ◽

Resource Setting ◽

Low Resource Setting ◽

Epithelioid Trophoblastic Tumor ◽

Β Hcg

Objective: To raise awareness of the existence of a rare type of malignant trophoblastic tumor and discuss the diagnostic challenges and management of this lesion in a low resource setting.Case report and intervention: A 35 -year -old G6P3 woman was referred to our facility on account of persistent vaginal bleeding due to a suspected incomplete miscarriage with a cervical mass. Her serum β-HCG was elevated (36,900 mIU/ml) and examination showed a bleeding cervical mass. An initial histopathological diagnosis of moderately differentiated squamous cell carcinoma was reviewed to epithelioid trophoblastic tumor resulting in an extra-fascial hysterectomy. A final histopathological diagnosis of hybrid Epithelioid Trophoblastic Tumor and Choriocarcinoma (ETT/CC) was made after external review and immunohistochemistry. She received subsequent chemotherapy.Conclusion: Epithelioid trophoblastic tumor and its hybrids are difficult to diagnose. They may be diagnosed as moderately differentiated squamous cell carcinoma especially in low resource settings where cervical squamous cell carcinoma is relatively more common. A high index of suspicion, a serum β HCG test and close collaborationbetween clinicians and pathologists can help make the diagnosis.Funding: NoneKeywords: Epithelioid trophoblastic tumour, choriocarcinoma, diagnostic challenges, low resource setting

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Epithelioid trophoblastic tumor coexisting with choriocarcinoma around an abdominal wall cesarean scar: a case report and review of the literature

Journal of Medical Case Reports ◽

10.1186/s13256-020-02485-8 ◽

2020 ◽

Vol 14 (1) ◽

Author(s):

Chunfeng Yang ◽

Jianqi Li ◽

Yuanyuan Zhang ◽

Hanzhen Xiong ◽

Xiujie Sheng

Keyword(s):

Abdominal Wall ◽

Chorionic Gonadotropin ◽

Human Chorionic Gonadotropin ◽

Unknown Origin ◽

Exploratory Laparotomy ◽

Cesarean Scar ◽

Trophoblastic Tumor ◽

Epithelioid Trophoblastic Tumor ◽

Trophoblastic Tumors ◽

Wall Lesion

Abstract Background Mixed gestational trophoblastic neoplasms are extremely rare and comprise a group of fetal trophoblastic tumors including choriocarcinomas, epithelioid trophoblastic tumors, and placental site trophoblastic tumors. We present a case of a patient with extrauterine mixed gestational trophoblastic neoplasm adjacent to the abdominal wall cesarean scar. On the basis of a literature review, this type of case has never been reported before due to the unique lesion location and low incidence. Case presentation Our patient was a 39-year-old Chinese woman who had a history of two cesarean sections and one miscarriage. She had a recurrent anterior abdominal wall mass around her cesarean scar, and the mass was initially suspected of being choriocarcinoma of unknown origin. The patient had concomitant negative or mildly increased serum β-human chorionic gonadotropin at follow-up and no abnormal vaginal bleeding or abdominal pain. However, she underwent local excision twice and had two courses of chemotherapy with an etoposide and cisplatin regimen. She finally opted for exploratory laparotomy with abdominal wall lesion removal, subtotal hysterectomy, bilateral salpingectomy, and left ovarian cyst resection, which showed the abdominal wall lesion, whose components were revealed by microscopy and immunohistochemical staining to be approximately 90% epithelioid trophoblastic tumors and 10% choriocarcinomas from a solely extrauterine mixed gestational trophoblastic neoplasm around an abdominal wall cesarean scar. Conclusions It is worth noting whether epithelioid trophoblastic tumor exists in the setting of persistent positive low-level β-human chorionic gonadotropin. More studies are required to provide mechanistic insights into these mixed gestational trophoblastic neoplasms.

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Ovarian Dysgerminoma in Pregnant Women with Viable Fetus: A Rare Case Report

Case Reports in Oncology ◽

10.1159/000513622 ◽

2021 ◽

pp. 141-146

Author(s):

Reda Youssef ◽

Gamal Sayed Ahmed ◽

Samir Alhyassat ◽

Sanaa Badr ◽

Ahmed Sabry ◽

...

Keyword(s):

Gestational Age ◽

Ovarian Neoplasms ◽

Imaging Modality ◽

Reproductive Age ◽

Diagnostic Challenge ◽

Short Period ◽

Ovarian Dysgerminoma ◽

The Impact ◽

In Pregnancy

Dysgerminoma is an uncommon malignant tumor arising from the germ cells of the ovary. Its association with pregnancy is extremely rare, with a reported incidence of about 0.2–1 per 100,000 pregnancies. Women in the reproductive age group are more commonly affected. It can be extremely rare to conceive naturally, without assisted reproductive interventions, in cases with ovarian dysgerminoma. If a pregnancy does occur with a concurrent dysgerminoma, it is even more unusual to carry the pregnancy to viability or childbirth without fetal or maternal compromise. We report a case of right ovarian dysgerminoma in a young female with a viable intrauterine pregnancy at 10 weeks, which is rarely diagnosed and managed at this gestational age. Numerous factors played a role in her favorable outcome, including early suspicion by ultrasound and presenting history, surgery, histopathological assessment, imaging, and involvement of the multidisciplinary oncology team. Ovarian neoplasms may rapidly increase in size within a short period with little or no symptoms. This poses a diagnostic challenge for obstetricians and oncologists. Hence, we aimed to evaluate the role of imaging in pregnancy using ultrasound as an imaging modality for both early detection of ovarian neoplasms and for follow-up. In conclusion, patients with ovarian dysgerminoma in pregnancy can have favorable outcomes. Treatment should be individualized on a case-to-case basis, depending on many factors; cancer stage, previous reproductive history, the impact of imaging in staging or follow-up of tumor on the fetus, fetal gestational age, and whether termination of the pregnancy can improve survival or morbidity for the mother.

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Benign Osteoblastoma Involving Maxilla: A Case Report and Review of the Literature

Case Reports in Dentistry ◽

10.1155/2012/351241 ◽

2012 ◽

Vol 2012 ◽

pp. 1-4 ◽

Cited By ~ 8

Author(s):

K. Bokhari ◽

M. S. Hameed ◽

M. Ajmal ◽

Rafi A. Togoo

Keyword(s):

Case Report ◽

Surgical Excision ◽

Good Prognosis ◽

Histopathological Diagnosis ◽

Diagnostic Challenge ◽

Diagnostic Dilemma ◽

Long Term Follow Up ◽

Rare Benign Tumor ◽

Benign Osteoblastoma ◽

Maxilla And Mandible

Background. Osteoblastoma is a rare benign tumor. This tumor is characterized by osteoid and bone formation with the presence of numerous osteoblasts. The lesion is more frequently seen in long bones and rarely involves maxilla and mandible. Due to its clinical and histological similarity with other bone tumors such as osteoid osteoma and fibro-osseous lesions, osteoblastoma presents a diagnostic dilemma.Case Report. Very few cases of osteoblastomas involving maxillofacial region have been reported in the literature. This case report involves osteoblastoma involving right maxilla in an 18-year-old male patient. Following detailed clinical examination, radiological interpretation, and histopathological diagnosis, surgical excision was performed. The patient was followed up for a period of 3 years and was disease free.Summary and Conclusion. Benign osteoblastoma involving jaw bones is a rare tumor. There is a close resemblance of this tumor with other lesions such as fibro-osseous lesions and odontogenic tumors and thus faces a diagnostic challenge. Surgical excision with a long-term follow-up gives good prognosis to this lesion—Benign Osteoblastoma.

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