Kyu-Rae Kim

Imaging Findings of Complications and Unusual Manifestations of Ovarian Teratomas

Ovarian teratomas can be associated with various complications and demonstrate a wide spectrum of clinical and imaging features. The complications include torsion (16% of ovarian teratomas), rupture (1%-4%), malignant transformation (1%-2%), infection (1%), and autoimmune hemolytic anemia (<1%). These complications require different therapeutic strategies; therefore, timely and accurate diagnosis of these complications is important for optimal patient treatment. In cases of complicated ovarian teratomas, the clinical manifestations provide only limited information and often overlap with those of other diseases. Furthermore, ovarian teratomas may have unusual clinical and imaging manifestations, thereby leading to misdiagnosis. These unusual manifestations include immature teratomas, monodermal teratomas (struma ovarii), combination tumors and collision tumors containing teratomas, and mature cystic teratomas without demonstrable fat or with pure fatty components. To provide adequate treatment and prevent misdiagnosis, it is necessary to be familiar with the imaging findings of both the complications and the unusual manifestations of ovarian teratomas.

Long-term oncologic outcomes after fertility-sparing management using oral progestin for young women with endometrial cancer (KGOG 2002)

OBJECTIVE To analyse the long-term oncologic outcomes of a fertility-sparing management using oral progestin in young women with endometrial cancer. METHODS We analysed 148 patients (age≤40 years) with stage IA, grade 1, endometrioid adenocarcinoma of the uterus who underwent fertility-sparing management using daily oral medroxyprogesterone acetate (MPA) or megestrol acetate (MA). RESULTS 115 (77.7%) showed complete response (CR) to progestin treatment, and 35 (30.4%) of them experienced recurrence after median follow-up time of 66 months. The 5-year recurrence-free survival was 68% (95% confidence interval [CI], 58.5-76.9%). However, 33 patients (22.3%) who failed to achieve CR underwent definitive surgical management, and no one had recurrence after median follow-up time of 41 months. During progestin treatment and at the time of recurrence, no patient showed clinical progression of disease over stage IA. Body mass index (BMI) ≥25 kg/m(2) was the only significant factor associated with a failure to achieve CR (odds ratio [OR], 3.00; 95% CI, 1.35-6.66; P=0.007). Upon multivariate analysis, BMI≥25 kg/m(2) (OR, 2.14; 95% CI, 1.06-4.31; P=0.033) was significantly associated with a higher risk of recurrence and the use of MPA (compared to MA) (OR, 0.44; 95% CI, 0.22-0.88; P=0.021), maintenance treatment (OR, 0.22; 95% CI, 0.05-0.94; P=0.042) and pregnancy (OR, 0.25; 95% CI, 0.11-0.56; P=0.001) were significantly associated with a lower risk of recurrence. CONCLUSION Fertility-sparing management was highly effective and safe. BMI<25 kg/m(2), MPA (compared to MA), maintenance treatment and pregnancy were associated with higher possibility of long-term success.

Surgical staging and adjuvant chemotherapy in the management of patients with adult granulosa cell tumors of the ovary

OBJECTIVE To analyze the role of surgical staging and adjuvant chemotherapy in patients with adult type granulosa cell tumor (GCT) of the ovary. METHODS Patients were divided into those with early-stage (stages I-II, n=93) and advanced-stage (stages III-IV, n=13) GCT and analyzed separately in this retrospective study. RESULTS Of the 93 patients with early-stage GCT, 30 were completely staged and 25 underwent lymph node dissection. After surgery, 17 patients received adjuvant chemotherapy with bleomycin/etoposide/cisplatin (BEP). None had lymph node metastasis. Completely staged patients had no recurrence or deaths. However, recurrences were observed in 9 of 63 patients (14.3%) who did not undergo complete staging, with four (6.3%) dying due to disease. The 5-year disease-free survival (DFS) rates of groups with and without complete staging were 100% and 84%, respectively (P=0.037). Adjuvant chemotherapy was not significantly associated with DFS (P=0.193). All patients with advanced-stage GCT underwent optimal cytoreduction and received adjuvant chemotherapy with BEP. None of the 6 patients who completed 6 cycles of BEP had recurrence, whereas 5 of the 7 patients (71.4%) who received fewer than 6 cycles of BEP had recurrences and 3 (42.9%) died due to disease. The 5-year DFS rates of these two groups were 100% and 50%, respectively (P=0.022), with cycles of chemotherapy being the only significant factor for DFS in patients with advanced-stage GCT. CONCLUSIONS Complete surgical staging is recommended, but lymph node removal is not recommended for early-stage GCT. Optimal debulking followed by six cycles of BEP chemotherapy is recommended for advanced-stage GCT.

Lymphoepithelial cysts of the pancreas: CT and sonographic findings

Abstract. Two cases of rare lymphoepithelial cyst (LEC) of the pancreas are presented. Although the histogenesis of this lesion is not known, it can be histologically differentiated from other pancreatic and retropancreatic cysts. The importance of its recognition is in the distinction from cystic neoplasm of the pancreas.

Alpha-synuclein in gastric and colonic mucosa in Parkinson's disease: Limited role as a biomarker

Gastric and colonic alpha‐synuclein immunoreactivity has been reported in patients with Parkinsons disease (PD). However, enteric alpha‐synuclein also has been reported in healthy individuals.

Unfavorable prognosis of small cell neuroendocrine carcinoma of the uterine cervix: a retrospective matched case-control study.

Objectives: This study was a matched case-control study to analyze the clinical and pathological characteristics and the prognosis of patients with small cell neuroendocrine carcinoma in the uterine cervix. Patients and Methods: Thirty-two patients were treated for small cell neuroendocrine carcinoma of the cervix (SCNEC) at Asan Medical Center between January 1996 and June 2008. For a 1-to-2 matched case-control study, 64 patients with squamous cell carcinoma of the cervix (SCC) whose clinical stage and age are consistent with the SCNEC group were selected. Medical records were retrospectively reviewed to analyze and compare the clinical and pathological outcomes. Results: In the SCNEC group, the median age was 45 years, and early stage (stage IIA or below) was 75.0%. The postoperative adjuvant therapy was given more frequently in the SCNEC group. The recurrence rate was 59.4%, and lung, bone, and liver were common sites in the SCNEC group. Parametrial involvement and lymphovascular space invasion were risk factors in the SCNEC group, whereas lymph node metastasis is the risk factor in the SCC group. The progression-free survival and overall survival were significantly shorter in the SCNEC group than in the SCC group (16.9 and 30.6 months vs 47.7 and 49.1 months, P < 0.05). Interestingly, survival outcomes in the early stage of SCNEC were remarkably worse than those of SCC and almost identical to those of the advanced stage of SCNEC. Conclusion: We confirmed the unfavorable prognosis related to hematogenous metastasis in SCNEC. The treatment modality of early-stage SCNEC, which is radical hysterectomy followed by adjuvant therapy, needs to be reevaluated.

Pulmonary benign metastasizing leiomyoma associated with intravenous leiomyomatosis of the uterus: clinical behavior and genomic changes supporting a transportation theory.

Benign metastasizing leiomyoma is a rare lesion characterized by benign-appearing smooth muscle tumor most frequently involving the lung and usually associated with a benign leiomyoma or intravenous leiomyomatosis of the uterus. The pathogenetic mechanism of the tumor has not been clarified, but the possibilities including hormone-sensitive in situ proliferations of smooth muscle bundles, mechanical displacement or intravascular spread of preexisting benign uterine tumor tissue, and metastasized very low-grade uterine leiomyosarcoma have been proposed. We described a case of pulmonary benign metastasizing leiomyoma associated with a uterine intravenous leiomyomatosis in a 46-year-old woman with a result of comparative genomic hybridization study. The 2 lesions showed significantly overlapping, if not identical, complex genomic changes in the comparative genomic hybridization, suggesting that the 2 lesions are closely related to each other. Unresected pulmonary nodules were left untreated for 13 months after the hysterectomy and wedge biopsy of 3 pulmonary nodules to show no further growth, suggesting clinical behavior of nonmalignant tumor in our case. Benign metastasizing leiomyomas may comprise a heterogenous group of tumors in terms of their malignant potential and pathogenetic mechanism.

PURE NON-GESTATIONAL CHORIOCARCINOMA OF THE OVARY DIAGNOSED BY DNA POLYMORPHISM ANALYSIS

Pure primary ovarian choriocarcinoma is a rare condition that can be of gestational or non‐gestational origin. Non‐gestational choriocarcinoma has been found to be resistant to single‐agent chemotherapy and has a worse prognosis than gestational choriocarcinoma, but it is difficult to distinguish the two types by routine histological examination. Herein is reported a case of primary pure non‐gestational choriocarcinoma of the ovary in a 33‐year‐old nulligravid woman, as confirmed by DNA polymorphism analysis. All tested microsatellite markers had identical DNA profiles with the same allelic sizes between the tumor and the myometrium of the patient, who was homozygous for three markers (BAT26, BAT25 and D17S250) and heterozygous for four (D2S123, D18S57, DCC and D18S58), supporting non‐gestational origin. The patient has no evidence of disease 17 months after surgery and four cycles of combination chemotherapy. This case demonstrates the usefulness of DNA polymorphism analysis for the determination of the origin of extrauterine choriocarcinoma. Clinical relevance of this method needs to be further studied and substantiated.

Clinical relevance of gain-of-function mutations of p53 in high-grade serous ovarian carcinoma.

Purpose Inactivation of TP53, which occurs predominantly by missense mutations in exons 4–9, is a major genetic alteration in a subset of human cancer. In spite of growing evidence that gain-of-function (GOF) mutations of p53 also have oncogenic activity, little is known about the clinical relevance of these mutations. Methods The clinicopathological features of high-grade serous ovarian carcinoma (HGS-OvCa) patients with GOF p53 mutations were evaluated according to a comprehensive somatic mutation profile comprised of whole exome sequencing, mRNA expression, and protein expression profiles obtained from the Cancer Genome Atlas (TCGA). Results Patients with a mutant p53 protein (mutp53) with a GOF mutation showed higher p53 mRNA and protein expression levels than patients with p53 mutation with no evidence of GOF (NE-GOF). GOF mutations were more likely to occur within mutational hotspots, and at CpG sites, and resulted in mutp53 with higher functional severity (FS) scores. Clinically, patients with GOF mutations showed a higher frequency of platinum resistance (22/58, 37.9%) than patients with NE-GOF mutations (12/56, 21.4%) (p=0.054). Furthermore, patients with GOF mutations were more likely to develop distant metastasis (36/55, 65.5%) than local recurrence (19/55, 34.5%), whereas patients with NE-GOF mutations showed a higher frequency of locoregional recurrence (26/47, 55.3%) than distant metastasis (21/47, 44.7%) (p=0.035). There were no differences in overall or progression-free survival between patients with GOF or NE-GOF mutp53. Conclusion This study demonstrates that patient with GOF mutp53 is characterized by a greater likelihood of platinum treatment resistance and distant metastatic properties in HGS-OvCa.

Micropapillary pattern in serous borderline ovarian tumors: Does it matter?

OBJECTIVE To evaluate the clinical and prognostic impact of micropapillary pattern in patients with serous borderline ovarian tumors (SBOT). METHODS We retrospectively assessed 130 consecutive patients with typical (n=97, 74.6%) or micropapillary (n=33, 25.4%) SBOT. Clinicopathologic factors and outcomes were compared between these two groups. RESULTS There were no significant between-group differences in age, menopausal status, parity, body mass index, cancer antigen 125 concentration, tumor size, tumor rupture, positive cytology, ovarian surface involvement, retrieved lymph nodes, use of laparoscopy, fertility-sparing and ovary-sparing procedures, complete staging and restaging, and adjuvant chemotherapy. However, the incidences of advanced stage (II-III) tumors (10.3% vs 36.4%, P=0.001), microinvasion (2.1% vs 15.2%, P=0.012), peritoneal implants (8.3% vs 33.4%, P<0.001), and lymph node involvement (0% vs 21.2%, P<0.001) were significantly higher in patients with micropapillary than with typical SBOT. Five patients with typical (5.2%) and three with micropapillary (9.1%) SBOT had recurrent disease (P=0.418), and one patient (3%) in micropapillary SBOT group died due to the disease (P=0.254). The 5-year disease-free survival (DFS) rates for patients with typical and micropapillary SBOT were 96% and 86%, respectively (P=0.148). All three patients with micropapillary SBOT who had recurrence had peritoneal implants (one noninvasive and two invasive). Multivariate analysis showed that peritoneal implant was the only significant factor related to DFS (P=0.002). CONCLUSIONS Because micropapillary SBOT was significantly associated with peritoneal implants, especially invasive implants, and lymph node involvement, complete staging procedures, including lymph node dissection, are recommended. However, micropapillary SBOT itself was not significantly associated with DFS. Peritoneal implant was the only factor independently associated with tumor recurrence.