Differential diagnosis of non-molar gestational trophoblastic neoplasia with ectopic pregnancy by clinical-pathological features.

PURPOSE: This study was presented to investigate the clinical-pathological characteristics of gestational trophoblastic neoplasia (GTN) following non-molar pregnancy and differentiated with ectopic pregnancy (EP). METHODS: The clinical data of 83 patients who were admitted for suspected GTN after non-molar pregnancy at the Women's Hospital School of Medicine Zhejiang University from January 2015 to September 2022 were selected for analysis. RESULTS: In total, 41 cases were confirmed non-molar GTN, including 31 choriocarcinoma, 9 PSTT (placental site trophoblastic tumor), and 1 ETT (epithelioid trophoblastic tumor), while 42 cases were confirmed EP. Compared with ectopic pregnancy, non-molar GTN patients had lower levels of serum progesterone compared with EP (3.81 nmol/L vs 17.70 nmol/L, P = 0.001). Based on the ultrasound, the thickness of the endometrium was thinner in patients with non-molar GTN compared with EP (0.565 cm vs 0.70 cm, P = 0.018). By histopathologic examination, the endothelium of non-molar GTN showed less decidual-like changes compared with EP (64.3% vs 14.6%, P = 0.001). CONCLUSION: A combination of serum progesterone levels, endometrium thickness, and histopathologic features of the endometrium can help to differentiate non-molar GTN and EP. Surgeries including hysteroscopy with curettage and/or laparoscopy are needed.

Retrospective analysis of clinical features and fertility outcomes with fertility-sparing treatment of placental site trophoblastic tumor.

OBJECTIVE: To analyze the methods, feasibility, efficiency, and fertility outcomes of fertility-sparing treatment for patients with placental site trophoblastic tumor (PSTT). METHODS: Clinical data of patients diagnosed with PSTT between April 1998 and April 2020 from Peking Union Medical College Hospital (PUMCH) were retrospectively collected. The clinical features, treatment, and outcomes of patients received fertility-sparing treatment were analyzed and compared with patients suffered hysterectomy. RESULTS: In total, 126 patients were included in the study and 29 of them received fertility-sparing treatment. Besides significantly younger age and lower proportion of antecedent term delivery were seen in fertility-sparing group than hysterectomy group, no significant differences were observed in stage, serum ß-hCG level, or interval from antecedent pregnancy between the two groups. Conservative surgery was selected individualized and none of them suffered salvage hysterectomy. Patients with clinical or pathological high-risk factors received adjuvant chemotherapy, yet the fertility-sparing treatment did not significantly lengthen chemotherapy duration. All patients in fertility-sparing group achieved complete remission without relapse after 36 to 176 months of follow-up and had sixteen healthy term delivery more than one year after the treatment. CONCLUSIONS: Fertility-sparing treatment for PSTT can be considered for young patients with localized uterine lesions who strongly desire to preserve their fertility potential. With individualized conservative surgery and selected adjuvant chemotherapy, fertility-sparing treatment will not influence the risk of relapse or overall survival and patients will achieve favorable pregnancy and live birth outcomes.

Placental site trophoblastic tumor (PSTT): a case report and review of the literature.

Placental site trophoblastic tumor (PSTT), also known as atypical choriocarcinoma, syncytioma, chorioepitheliosis or trophoblastic pseudotumor, is a rare gestational trophoblastic disease (0.25-5% of all trophoblastic tumors) and it is composed by neoplastic proliferation of intermediate trophoblasts at placental implantation site. It consists of aggregates or sheets of large, polyhedral to round, predominantly mononucleated cells with a characteristic vascular and myometrial invasion. Main differential diagnoses are gestational choriocarcinoma (GC) and epitelioid trophoblastic tumor (ETT). We present a case of PSTT in a 25-year-old woman. Neoplastic cells showed moderate/high nuclear pleomorphism, abundant amphophilic, eosinophilic and clear cytoplasm, numerous mitotic figures (10 mitoses/10 HPF), and myometrial invasion. Other features are necrosis, vascular invasion with replacement of myometrial vessels by tumor cells and hemorrhage. The patient showed typical low serum ß-hCG levels and high serum humane placental lactogen (hPL) levels.

Outcomes and prognostic factors of placental-site trophoblastic tumor: a retrospective study of 58 cases.

PURPOSE: Our goal was to assess the outcomes and explore the prognostic factors for patients with placental-site trophoblastic tumor (PSTT) through this retrospective analysis. METHODS: 2043 patients with gestational trophoblastic neoplasia (GTN) were registered at two tertiary hospitals between January 2003 and March 2021, of whom 58 (2.8%) were diagnosed with PSTT. We retrospectively analyzed the clinico-pathological characteristics, treatments, outcomes and prognostic factors. RESULTS: Only 4 patients died and 5 patients experienced a recurrence. Patients (n = 49) with stage I disease had a favorable prognosis, surgery with (n = 21) or without (n = 28) chemotherapy made no significant difference in overall survival (OS) (p = 0.251) or disease-free survival (DFS) (p = 0.425). 3 patients with stage I had fertility preserving surgery and successful pregnancy was achieved in 2 of them. The outcome of patients with advanced disease was poor. Univariate analysis revealed serum ß-hCG levels at diagnosis, FIGO stage IV and metastatic disease were significant predictors of both overall survival and disease-free survival. However, multivariate analysis indicated stage IV was the only significant independent predictor of adverse OS, while metastatic disease was the only significant independent predictor of adverse DFS. CONCLUSION: Surgery alone is sufficient for patients with stage I disease without high-risk factors. The prognosis of patients with advanced stage disease remains poor. Stage IV and metastatic disease were the most critical risk factors.

"It's not lupus". A placental site trophoblastic tumor presenting as a lupus-like paraneoplastic syndrome. A grand round case.

Introduction: Placental site trophoblastic tumor (PSTT) is a rare subtype of gestational trophoblastic disease. Association of PSTT and nephrotic syndrome is exceedingly rare and has been described in 8 cases thus far. In all cases hysterectomy was performed within months after onset of symptoms, leading to immediate remission of nephrotic syndrome, except for one patient who died of complications of PSTT. Case: We describe the history of a woman in which PSTT was discovered years after onset of nephrotic syndrome. Kidney biopsy revealed lupus-like mesangiocapillary nephritis and over time the patient developed additional symptoms mimicking systemic lupus erythematosus (SLE). Discussion: We provide an overview of the literature on this clinical entity and elaborate on its pathophysiology. In addition, we reflect on the phenomenon of anchoring bias, that led physicians to assume the patient had SLE without questioning this diagnosis in the light of the unexplained finding of increased tumor markers.

Placental site trophoblastic tumor in early stage: a case report.

Here, the authors present a case of a placental site trophoblastic tumor (PSTT) in a 28-year-old gravida 1 para 1 living 1 woman three months after vaginal delivery of a female infant at diagnosis in 2014. The patient was FIGO Stage I and finally underwent a total laparoscopic hysterectomy with ovarian conservation. Subsequently, the patient received two cycles of EMA/CO chemotherapy. Patient is on regular follow-up (clinical exam, ß-hCG tests, pelvic and abdominal sonography) and has shown no signs of local or systemic recurrence for 24 months.

Placental Site Trophoblastic Tumor: A Distinct Entity of Gestational Trophoblastic Disease Experience from a Tertiary Referral Center in Hong Kong.

OBJECTIVE: To review the clinical and pathological characteristics of patients with placental site trophoblastic tumor (PS TT) managed in a tertiary referral center in Hong Kong. STUDY DESIGN: Patients with a diagnosis of PSTT from 1995 to 2012 were identified from a computer database. Clinical and patho- logical data were obtained from medical records and the electronic database. RESULTS: Ten patients with PSTT were identified. Only 4 patients (40%) had disease confined to the uterus at presentation (Stage I). The most common site of metastasis was the lung. Four patients had pretreatment serum hCG levels <1,000 IU/L, and all of them had disease 'confined to the uterus. Of the 4 patients with Stage I disease 3 had hysterectomy only and 1 had both hysterectomy and chemotherapy. All 4 patients achieved complete remission; although 1 of them had a recurrence successfully treated with che- motherapy. For patients with Stage III/IV disease most of them had both hysterectomy and chemotherapy. Only 1 patient (20%) was alive without evidence of disease. CONCLUSION: Patients with Stage I disease have excellent prognosis after hysterectomy, and adjuvant treatment is not recommended. A low pretreatment serum hCG level (<1,000 IU/L) was a good predictor of early stage disease. The prognosis for patients with metastatic disease was poor despite surgery and com- bination chemotherapy.

[Placental site trophoblastic tumor with atypical choriocarcinoma. Case Report]. / Tumor trofoblástico del sitio placentario con evolución a coriocarcinoma. Caso clínico.

BACKGROUND: The placental site trophoblastic tumor is the second rarest tumor of trophoblastic disease, with less than 300 cases reported. Clinical presentation it's a diagnostic challenge, it has an unpredictable malignant potential, and it can develop choriocarcinoma and even have simultaneous presentation. CASE REPORT: We present the case of a 34 year old woman with chronic transvaginal bleeding since her last cesarean (five months ago), with histologic diagnosis of placental site trophoblastic tumor and inmuno- histoquimic report of choriocarcinoma, this early diagnosis improves her survival prognostic. CONCLUSION: placental site trophoblastic tumor and their evolution to choriocarcinoma have a low rate but it's possible. That it's the reason why complete diagnosis with biopsy, histologic and inmunohistoquimic report is mandatory to improve therapeutic.

Placental site trophoblastic tumor: report of a tertiary center experience.

OBJECTIVE: To analyze the clinical and pathological characteristics of placental site trophoblastic tumor (PSTT) cases and to discuss the diagnosis, treatment, and prognosis of PSTT. MATERIALS AND METHODS: The clinical and pathological data of eight patients with PSTT at Istanbul Medical Faculty Hospital from 1988 to 2010 were analyzed retrospectively. RESULTS: The mean age of the patients was 31 years. The antecedent pregnancy was full-term delivery in most of the patients (6/8, 75%). The mean interval from last pregnancy to diagnosis of PSTT was 35 months (range, six to 192). Serum human chorionic gonadotropin (hCG) levels at the time of diagnosis ranged from 0.1 to 2280 mIU/ml (mean, 614). All patients had Stage 1 disease and ultimately underwent hysterectomy. None of the patients received adjuvant chemotherapy. One patient died of an unknown reason, one month after the surgery. The rest of the patients were alive and without evidence of disease after an average of 3.5 years (range, one to 11) of follow-up. CONCLUSION: Hysterectomy alone can provide long-term survival in early-stage disease.

Placental-site trophoblastic tumor and fluorine-18-fluorodeoxyglucose positron emission tomography/computed tomography.

OBJECTIVE: Pre-operative imaging characteristics of placental site trophoblastic tumor (PSTT) are variable and non-specific. Although magnetic resonance imaging (MRI), ultrasonography, chest CT and X-rays findings have been studied the fluorine-18-fluorodeoxyglucose positron emission tomography/computed tomography (¹8F-FDG PET/CT) findings of PSTT have not been previously documented. We present the findings of a first case of PSTT evaluated by pre-operative ¹8F-FDG PET/CT. A suspicious mass was biopsied and revealed PSTT in post-operative pathological examination. She was referred to the gynecology-oncology department. The ¹8F-FDG PET/CT scan revealed a 27 x 20 mm laterally expanded lesion that showed increased ¹8F-FDG uptake (SUVmax: 5.20) on the right isthmus of the uterus. The ¹8F-FDG PET/CT findings were in accordance with those from chest X-ray/s, CT and pelvic ultrasonography. A systematic, nerve sparing, paraaortic and pelvic lymph node dissection along with total hysterectomy and salpingoopherectomy was performed. The patient was discharged uneventfully. CONCLUSION: ¹8F-FDG PET/CT scan was able to identify the mass in the uterus which was shown by pathology to be PSTT. This finding of PET/CT was in accordance with other imaging techniques. Lymphatic mapping of ¹8F-FDG PET/CT in this case was also in accordance with surgery and pathology findings.

[Value of laparoscopic surgery in the diagnosis of suspected gestational trophoblastic neoplasia cases with uterine mass].

OBJECTIVE: To evaluate the value of laparoscopic surgery in the diagnosis of suspected gestational trophoblastic neoplasia (GTN) cases with uterine mass. METHODS: The clinical characteristics of patients underwent laparoscopic surgery for a suspected diagnosis of GTN with uterine mass in Peking Union Medical College Hospital from November 2009 to November 2014 were retrospectively reviewed and analyzed. GTN and other pregnant-related disease were definitely diagnosed by pathological findings. The prognoses of the GTN cases were also investigated. RESULTS: Sixty-two patients with a suspected diagnosis of GTN with uterine mass were studied. Among them, 17 cases were definitely diagnosed as GTN, including 8 choriocarcinoma, 5 invasive mole and 4 placental site trophoblastic tumor (PSTT). The other 45 cases were diagnosed as benign pregnancy-related diseases, including 29 cornual pregnancy, 6 cesarean scar pregnancy, 5 placenta accreta, 4 intramural uterine pregnancy and 1 exaggerated placental site. There were no significantly differences between the two groups in average age, preoperative value or tendency of ß-hCG, and location or size of lesions (P>0.05). More GTN patients showed a history of hydatidiform mole [5/17 vs 4% (2/45) , P>0.05], and more patients with benign pregnancy-related disease showed a history of cesarean section [38% (17/45) vs 1/17, P>0.05]. No serious perioperative complication was found in these patients received laparoscopic surgery. All GTN patients achieved complete remission by chemotherapy later. Except for 1 case loss, no recurrence was found in 11 low-risk stage I cases with an average follow-up period of 11- 66 months, 1 high-risk stage I case with a follow-up period of 61 months and 4 cases PSTT with a follow-up period of 13-66 months. CONCLUSIONS: There were some atypical GTN cases with uterine mass, which were difficult to be differentiated from benign pregnancy-related diseases according to the clinical characteristics. Laparoscopic surgery with a pathologic diagnosis could be an essential way with efficiency and safety.

Placental site trophoblastic tumor on endometrial polyp: a case report.

Placental site trophoblastic tumor (PSTT) is the least common form of gestational trophoblastic disease (GTD), and is biologically different from other forms of GTD. There is a wide clinical spectrum of presentation and behavior ranging from a benign condition to an aggressive disease with a fatal outcome. The authors document a case of PSTT on an endometrial polyp. A 51-year-old woman had abnormal vaginal bleeding for the duration of two months. Her past history included a vaginal delivery in 1998. Her physical examination was normal. Tumor markers were at normal levels. Serum beta- human chorionic gonadotropin (hCG) level was 19 mIU/ml and human placental lactogen (hPL) level was in the normal range. The patient underwent an operative hysteroscopy. On examination the uterine cavity appeared to be occupied by a pedunculated polypoid neoformation measuring about 2.5 cm in diameter which was removed and later determined to be a PSTT. There were occasional mitotic figures (0-1/10 high power field). The patient underwent hysterectomy and bilateral salpingo-oophorectomy. The patient has no evidence of disease six months after surgery. The authors conclude that a high mitotic count and atypical undifferentiated pathological features are significant poor prognostic factors for survival in PSTT. Hysterectomy represents the gold standard of treatment in all cases of disease confined to the uterus.

Exaggerated placental site/placental site trophoblastic tumor: an underestimated risk factor for emergency peripartum hysterectomy.

OBJECTIVE: To assess the indications and possible underlying causes of emergency peripartum hysterectomy (EPH) at the present hos- pital during the 2001-2011 period. MATERIALS AND METHODS: A revision of the charts and pathology reports corresponding to 42,728 parturients. RESULTS: During the study period, 25 peripartum hysterectomies were performed (0.61/1.000), of which 23 were EPHs (0.54/1.000) and two were planned cesarean hysterectomies. The indication for EPH was acute postpartum hemorrhage in 22 of 23 instances (95.7%). Roughly two-thirds of the operated uteri (16/25, 64%) showed placental site anomalies, half corresponding to different degrees of placental accretism and half to anomalies derived from the implantation site intermediate trophoblast. In five cases (31%), the anomaly was an exaggerated placental site and three cases corresponded to placental site trophoblastic tumors. Of the 16 cases showing placental site anomalies, ten (62.5%) were associated with one or more previous cesarean sections. CONCLUSIONS: Roughly one-third of EPHs performed at the present center during the last ten years were associated with placental site anomalies originating in the implantation site intermediate trophoblast (exaggerated placental site and placental site trophoblastic tumor). This association has not been described before, and should be taken into consideration when facing acute peripartum hemorrhage predisposing to EPH.

Attempted conservative management of a placental site trophoblastic tumor: a case report.

BACKGROUND: Placental site trophoblastic tumors (PSTTs) are rare malignant forms of gestational trophoblastic neoplasia (GTN). Controversy exists regarding the most important pathologic or radiologic predictors of extent of disease. Consequently, there is limited information as to the best candidates for conservative surgery. CASE: A 28-year-old female presented 18 months after a term delivery with a biopsy confirmed PSTT. She declined hysterectomy. Imaging revealed a locally limited lesion without myometrial invasion, and no evidence of metastatic disease. She was given two cycles of neoadjuvant etoposide, methrotrexate, actinomycin D, cyclophosphamide, and vincristine (EMA/CO) chemotherapy followed by an attempt at laparoscopically guided hysteroscopic resection. Pathology showed extensive myometrial invasion with positive surgical margin, and our recommendation for hysterectomy with pelvic lymph node dissection was accepted. Postoperatively, she was given two cycles ofpaclitaxel, cisplatin alternating with paclitaxel, etoposide (TP/TE) chemotherapy. CONCLUSION: Fertility sparing options are desirable and should be considered. However, as our case and much of the literature demonstrates, hysterectomy remains the most successful treatment.

[Reservation of fertility for seventeen patients with placental site trophoblastic tumor].

OBJECTIVE: To approach the efficiency and feasibility of preserving the fertility for patients with placental site trophoblastic tumor (PSTT). METHODS: Totally 2 086 cases of gestational trophoblastic neoplasm (GTN) patients registered in Peking Union Medical College Hospital between 1998 and 2013. Fifty-seven of them were PSTT patients, 40 cases of which suffered hysterectomy, the rest 17 PSTT patients who preserved their fertility were analyzed retrospectively. The computerized database of clinical and pathological reports was reviewed in this cohort. RESULTS: The clinical manifestation of PSTT was not specific compared to other types of GTN. The average age of the 17 patients was 29.5 years old (range 22-39 years). The most common antecedent pregnancy was term birth (8 cases), the others were spontaneous abortion in 4 case, artificial abortion in 3 cases and molar pregnancy in 2 cases. The baseline serum ß-hCG was slightly elevated and 12 patients (12/15) were less than 1 000 U/L. In this cohort, nine of the patients were in stage I, while the other eight cases were in stage III . The patients suffered conservative surgery, including dilation and curettage of uterus in 7 cases, open abdomen uterine lesion excision in 4 cases, laparoscopic uterine lesion excision in 3 cases, hysteroscopic uterine lesion excision in 1 case, and pulmonary lobectomy in 2 cases. Two of the patients didn't received chemotherapy, while the other 15 cases suffered combination chemotherapy. Compared with 40 patients who suffered hysterectomy during the same interval, fertility preservation group did not result in poor outcomes or high risk of relapse rate. Six subsequent pregnancies happened after the therapy, two of them were during their second-trimester, while four patients had healthy babies by vaginal delivery in two and cesarean section in two. The scar of the uterus was fairly well during the cesarean sections. CONCLUSIONS: Reservation of fertility therapy could be considered in highly-selected patients for young women who strongly desired to preserve their fertility and with localized lesion. Exactitude follow-up after therapy should be recommended. Contraception should also be recommended for at least one year after the chemotherapy. Vaginal delivery could be an option for the future pregnancies.

Ovarian non-gestational placental site trophoblastic tumor with lung metastasis: further evidence for a distinct category of trophoblastic neoplasm.

We previously described a series of cases which characterize a distinct group of primary ovarian placental site trophoblastic tumor (PSTT) and epithelioid trophoblastic tumor (ETT) as a non-gestational set consistent with germ cell type/origin. Here we report a new case of ovarian non-gestational PSTT. The patient was a 13 year-old young female admitted for a spontaneous pneumothorax of the left lung. The pathology of lung wedge excision specimen demonstrated metastatic PSTT and ovarian biopsy showed atypical intermediate trophoblastic proliferation which was found to be PSTT in the subsequent salpingo-oophorectomy specimen. In the ovary, the tumor was composed of singly dispersed or small clusters of predominantly mononuclear cells and rare multinucleated cells extensively infiltrating the ovarian parenchyma, tubal mucosa, and paraovarian/paratubal soft tissue. A minor component of mature cystic teratoma (less than 5% of total tumor volume) was present. Immunohistochemically, the neoplastic cells of main tumor were diffusely immunoreactive for hPL, Gata3 and AE1/AE3, and had only rare hCG-positive or p63-positive cells. The morphology and immunohistochemical results support a PSTT. Molecular genotyping revealed an identical genotype pattern between the normal lung tissue and the metastatic PSTT, indicating its non-gestational nature of germ cell type/origin. This case represents the first case of such tumor with distant (lung) metastasis. This case also provides further evidence to support our recommendation that primary ovarian non-gestational intermediate trophoblastic tumors of germ cell type/origin, including PSTT and ETT, should be formally recognized in classification systems.

Placental site and epithelioid trophoblastic tumours: diagnostic pitfalls.

OBJECTIVE: To describe the clinical and histological pitfalls in the diagnosis of placental site trophoblastic tumours (PSTT) and epithelioid trophoblastic tumours (ETT), two rare types of gestational trophoblastic neoplasia (GTN). METHODS: This retrospective, observational, study was carried out in the French Trophoblastic Disease Reference Centre, Lyon, between 2000 and 2011. Due to the many similarities in the diagnosis, treatment and prognosis of PSTT and ETT, these two types of tumour were investigated together. Twenty-two patients with PSTT or ETT were analysed. RESULTS: The clinical presentation of these two types of tumour was irregular vaginal bleeding (55%) or amenorrhoea (27%), with a median plasma hCG level of 205IU/L. Seven of the 22 patients (32%) were initially misdiagnosed as an ectopic pregnancy. Median age at presentation was 35-years, with a median interval of 12months between the antecedent pregnancy and diagnosis of PSTT or ETT. The initial histological diagnosis was incorrect in 7/18 (39%) patients; there was a major disagreement with the referral pathologist in five of these seven patients (28%). CONCLUSIONS: PSTT and ETT are the most difficult types of GTN to diagnose clinically and histologically. An incorrect diagnosis can lead to significant therapeutic deviations from the recommended first-line treatment, namely hysterectomy. Clinical and histological expertise is essential to avoid the pitfalls in the diagnosis of PSTT and ETT.

Long-term outcome of placental site trophoblastic tumor in The Netherlands.

OBJECTIVE: To evaluate the outcome of 16 cases of placental site trophoblastic tumors (PSTTs) treated throughout The Netherlands. STUDY DESIGN: Patients with PSTT between 1991 and 2009 were identified using the nationwide network and registry of histopathology and cytopathology in The Netherlands (PALGA) and medical records. RESULTS: Data for 16 patients could be retrieved. The median age of the patients was 32 years. In 7 cases the antecedent pregnancy was a miscarriage and in 6, a term delivery. Clinical data on 3 patients could not be retrieved. Six patients were low-risk according to the International Federation of Gynecology and Obstetrics staging system. The median human chorionic gonadotropin (hCG) level was 46 IU/L, but in 4 patients the hCG level was not elevated. In the majority of patients a hysterectomy was performed, and 5 patients needed additional chemotherapy. There was only 1 recurrence and there were no fatalities. CONCLUSION: This study emphasizes the need for an international registration. No fatalities were registered. Because of the low incidence and limited experience of general gynecologists with this disease, there is a preference for centralization of all patients with PSTT regardless of their stage.

Placental Site Trophoblastic Tumor in Mediastinal Recurrence of Mixed Germ Cell Tumor of the Testis: Report of a Case and Review of the Literature.

We present a rare case of a 42-year-old man diagnosed with a placental site trophoblastic tumor in combination with teratoma in a mediastinal recurrence of a testicular germ cell tumor post-orchiectomy and chemotherapy. To the best of our knowledge, this is the eighth case of placental site trophoblastic tumor in a male reported so far in the English literature. The purpose of this case report is to add data to the existing literature, review the literature, discuss the differential diagnoses with emphasis on morphologic and immunohistochemical differences between trophoblastic tumors, and highlight the management implications of a correct diagnosis.

Placental site trophoblastic tumours and the concept of fertility preservation.

The standard management of placental site trophoblastic tumours (PSTTs) is a radical hysterectomy with pelvic lymph node sampling. We present five cases to demonstrate a modified Strassman procedure (MSP), which is an alternative fertility-sparing technique. Each had a presumed solitary uterine PSTT. Following surgery, one patient remained in remission with her fertility intact. The other four underwent a completion hysterectomy because of incomplete excision of the disease. No residual disease was later found in two of these four uteri. This treatment should only be offered after extensive counselling. We intend to investigate the use of intraoperative frozen section analysis with cold-knife dissection in future.