Erdheim-Chester disease (ECD) is a rare type of non-Langerhans cell histiocytosis and is characterized by the diffuse histiocytic infiltration of multiple organs. Retroperitoneal ECD, especially with uterine involvement, is extremely rare. We report about a 73-year-old woman who presented with vaginal spotting for a month and experienced abdominal pain along with pus-like urine. Computed tomography revealed an irregular mass-like lesion in the uterus, possibly a uterine sarcoma, invading the ureter, rectosigmoid, and bladder. A tissue biopsy of the retroperitoneal mass revealed typical morphological and immunohistochemical features of ECD. However, clinical features, especially long bone involvement, did not coincide with ECD, and BRAF V600E gene mutation was not detected. We made a diagnosis of atypical retroperitoneal ECD mimicking uterine sarcoma with multiple organ involvement.

Erdheim-Chester disease (ECD) is a rare type of non-Langerhans cell histiocytosis and is characterized by the diffuse histiocytic infiltration of multiple organs. Retroperitoneal ECD, especially with uterine involvement, is extremely rare. We report about a 73-year-old woman who presented with vaginal spotting for a month and experienced abdominal pain along with pus-like urine. Computed tomography revealed an irregular mass-like lesion in the uterus, possibly a uterine sarcoma, invading the ureter, rectosigmoid, and bladder. A tissue biopsy of the retroperitoneal mass revealed typical morphological and immunohistochemical features of ECD. However, clinical features, especially long bone involvement, did not coincide with ECD, and BRAF V600E gene mutation was not detected. We made a diagnosis of atypical retroperitoneal ECD mimicking uterine sarcoma with multiple organ involvement.

Indirect inguinal hernia containing an ovary is a rare condition, especially in adult women who do not have any other genital tract anomalies. In addition, inguinal hernia containing an ovary and endometriosis is exceedingly rare. In the present report, we describe a case of indirect inguinal hernia containing an ovary, fallopian tube, and endometriosis. Laparoscopic repair was performed successfully using polypropylene mesh for the treatment of the inguinal hernia.
Adult* ; Endometriosis* ; Fallopian Tubes* ; Female ; Hernia, Inguinal* ; Humans ; Ovary* ; Polypropylenes

Leiomyomatosis peritonealis disseminata (LPD) is a rare condition characterized by multiple subperitoneal nodules of benign smooth muscle. Wilson and Peale were the first to describe the multiple peritoneal leiomyomas, while Taubert et al. clearly delineated the features of the lesion and named it LPD. Approximately 100 cases of this disease have been reported in the world literature. High levels of exogenous and endogenous female gonadal steroids is associated with LPD, it suggests that estrogen and progesterone play important role in the pathogenesis of LPD. We report a case of leiomyomatosis peritonealis disseminata with review of literature.
Estrogens ; Female ; Gonads ; Humans ; Leiomyoma ; Leiomyomatosis* ; Muscle, Smooth ; Progesterone ; Steroids

Ovarian dermoid cysts are one of the most common benign neoplasms in women. Rectal fistula formation due to an ovarian dermoid cyst, particularly a benign dermoid cyst, is extremely rare. A 17-year-old girl with symptoms of lower abdominal pain, passage of sebaceous materials in the stool, and hematochezia was found to have an 11-cm dermoid cyst complicated with a rectal fistula formation. Laparoscopic repair of the rectal fistula was performed successfully with bilateral ovarian cystectomies. This case presents the rare formation of a fistula between a benign dermoid cyst and the rectum and its treatment using laparoscopic repair without laparotomy.
Abdominal Pain ; Adolescent ; Cystectomy ; Dermoid Cyst* ; Female ; Fistula ; Gastrointestinal Hemorrhage ; Humans ; Laparoscopy ; Laparotomy ; Rectal Fistula* ; Rectum

OBJECTIVE: This study was aimed to compare immunohistochemical expression of estrogen receptor (ER)-alpha, progesterone receptor (PR), and calponin in gonadotrophin-releasing hormone agonist (GnRH-a)-sensitive and -resistant uterine fibroids. METHODS: We collected data retrospectively. The sensitive group consisted of women who had reduction in uterine volume greater than 40% following GnRH-a treatment. Uterine volume was either reduced by less than 10%, or was increased in the resistant group. A tissue microarray was constructed using formalin-fixed, paraffin-embedded tissues, 31 and 26 patients for the sensitive and resistant groups, respectively. Tissue sections were immunostained with antibodies against ER-alpha, PR, and calponin. The intensity and area of the immunohistochemical reactions were evaluated using a semi-quantitative scoring system. The Mann-Whitney U-test, Fisher's exact test, and Spearman's rank correlation test were used for analysis of data. RESULTS: PR (P = 0.04) and calponin (P = 0.03) showed a significantly higher staining intensity in the resistant group than in the sensitive group. Both groups showed comparable expression of ER-alpha (P = 0.23). In correlation analysis between changes in uterine volume after GnRH-a therapy and clinicopathological factors, the immunohistochemical intensity of PR (P = 0.04) and calponin (P = 0.03) was significantly correlated with changes in uterine volume. CONCLUSION: This study shows that GnRH-a resistance of uterine fibroids is not related to ER-alpha content, but the expression of PR and calponin is related with GnRH-a resistance.
Antibodies ; Estrogens* ; Female ; Humans ; Leiomyoma* ; Progesterone* ; Receptors, Progesterone* ; Retrospective Studies

Bleeding due to cardiac perforation by endocardial pacemaker lead is a rare complication. We report one case of left hemothorax due to right ventricular perforation after the insertion of permanent transvenous pacemaker. Operative finding showed a pacing lead penetrating right ventricle, pericardium, and left pleura sequentially, but there was no evidence of hemopericardium.
Heart Ventricles ; Hemorrhage ; Hemothorax* ; Pacemaker, Artificial ; Pericardial Effusion ; Pericardium ; Pleura

PURPOSE: To assess the therapeutic value of two different drainage catheters in treating chronic subdural hematoma (CSDH). MATERIALS AND METHODS: Two types of drainage catheters can be used to treat CSDH according to the position of holes in the catheter: open-type or closed-type catheter. In this retrospective study, 199 total patients with CSDH were reviewed according to catheter type. Among them, 84 patients were and 113 in the closed-type group (holes positioned within the distal-most 1 cm of the catheter). The surgeon selected the catheter type. Total drainage volume, initial drainage volume within 2 days, percentage of initial drainage volume per total drainage volume, duration of catheter insertion, and reoperation rate were compared. RESULTS: Total drainage volume was not different between the two groups (p=0.333). The initial drainage volume within 2 days was larger in the open-type group than closed-type group (p=0.024), but the percentage of initial drainage volume per total drainage volume was not different (p=0.354). The duration of catheter insertion was shorter in the open-type group than closed-type group (p=0.015). The reoperation rate of CSDH was also higher in the open-type group than closed-type group (p=0.004). CONCLUSION: CSDH drainage with an open-type catheter is faster compared with a closed-type catheter, but total drainage volume is similar and reoperation rate is higher. Therefore, the open-type catheter for CSDH drainage has limited clinical value.
Adolescent ; Adult ; Aged ; Aged, 80 and over ; Catheters/*adverse effects ; Child ; Child, Preschool ; Drainage ; Equipment Design ; Female ; Hematoma, Subdural, Chronic/surgery/*therapy ; Humans ; Infant ; Male ; Middle Aged ; Retrospective Studies

OBJECTIVE: This study was designed to evaluate the survival benefit of laparoscopic surgical staging (LSS)-guided tailored radiation therapy (RT) in locally advanced cervical cancer (LACC). METHODS: We retrospectively reviewed 89 LACC patients' medical records who primarily received non-surgical treatment, of which pretreatment LSS was performed in 20 (LSS group) and primary chemoradiation therapy (CCRT) without LSS (CCRT group) was carried out in 69 from January 2000 to January 2006. We analyzed clinical characteristics, pretreatment imaging study results and survival outcomes including disease free survival (DFS) and overall survival (OS) to compare them between the two groups. RESULTS: There were as many as eight cases (40%) of LSS related complications. The mean time interval between LSS and RT or CCRT was 26.6 days (+/-18.8 days). Six out of twenty (30%) in LSS group and 10 out of 69 (14.5%) in CCRT group received extended field RT when paraaortic lymph nodes (LNs) were positive based on the pathologic findings after LSS and the results of imaging studies, respectively. Three-year DFS and OS were both better in 33 imaging-negative CCRT group patients than those in 4 imaging-negative/pathology-positive (false negative) patients after LSS (3-year DFS, 50% vs. 87%, p=0.022; 3-year OS, 50% vs. 84%, p=0.033). The 5-year DFS rates were 52% and 55% in LSS group and in CCRT group, respectively (p=0.28). The 5-year OS rates were 68% in LSS group and 62% in CCRT group without significant difference between the two groups (p=0.79). CONCLUSION: We found that LSS-based RT tailoring did not show survival benefit in LACC despite inaccuracy of imaging-based RT tailoring. Further studies are required to find new method to overcome this inaccuracy and improve survival outcomes.
Disease-Free Survival ; Humans ; Laparoscopy ; Lymph Nodes ; Medical Records ; Retrospective Studies ; Uterine Cervical Neoplasms

Adult granulosa cell tumors (AGCTs) presenting with massive ascites and elevated serum CA-125 levels have rarely been described in the literature. An ovarian mass, massive ascites, and elevated serum CA-125 levels in postmenopausal women generally suggest a malignant ovarian tumor, particularly advanced epithelial ovarian cancer. AGCT has low 18F-fluorodeoxyglucose uptake on positron emission tomography/computed tomography due to its low metabolic activity. In the present report, we describe a case of an AGCT with massive ascites, elevated serum CA-125 level, and low 18F-fluorodeoxyglucose uptake on positron emission tomography/computed tomography.
Adult* ; Ascites* ; Electrons* ; Female ; Granulosa Cell Tumor* ; Granulosa Cells* ; Humans ; Ovarian Neoplasms