Menstrual restoration in severe panhypopituitarism many years after cranial irradiation for suprasellar germinoma.

Purpose: We report a very rare case showing menstrual restoration in severe pan-hypopituitarism many years after cranial irradiation for suprasellar germinoma. Case: A 30-year-old, almost primarily amenorrheic woman with severe panhypopituitarism presented with cyclic genital bleeding for the previous five months. She had menstruated once, when she was 13 years old. When she was 14 years old, she was diagnosed with a suprasellar germinoma measuring 10 mm in diameter, which led to diabetes insipidus. Cranial irradiation with a total dose of 24 Gy and chemotherapy resulted in complete tumor remission. She developed severe hypopituitarism [luteinizing hormone (LH) = 0.4 mIU/mL, follicle-stimulating hormone (FSH) = 1.7 mIU/mL, and serum estradiol (E2) level < 10 pg/mL]. She had received multiple hormone replacement therapies for many years. When she was 29 years old, she expressed a desire to become pregnant. Serum gonadotropin and E2 levels increased (LH = 5.8 mIU/mL, FSH = 5.9 mIU/mL, and E2 = 58 pg/mL). She conceived with clomiphene therapy, and then delivered a healthy baby. Eight months after parturition, her basal body temperature and serum progesterone levels indicated recovery of ovulatory cycles. Ten months after parturition, she also spontaneously conceived. Conclusion: Menstrual restoration is very rare in severe panhypopituitarism after cranial irradiation. A relatively low dose of irradiation and small tumor size may have contributed to the recovery of menstruation in our patient.

Decreasing full-term neonatal birthweight over the past 2 decades in a single institute in Japan.

AIM: The aim of this study was to investigate whether full-term neonatal birthweight is decreasing or not. MATERIAL AND METHODS: Subjects were selected from the registration database of full-term singletons from a single local practitioner at two time-points, 1991 (n = 750) and 2011 (n = 442). Maternal age, parity, and smoking habits were recorded. Baseline characteristics included pre-pregnancy bodyweight (BW), body mass index (BMI), bodyweight before parturition, and weight gain during pregnancy. Neonatal birthweight and height were recorded. Variables correlated with neonatal size were investigated. RESULTS: Male birthweight decreased from 3248 ± 409 (n = 373) to 3149 ± 430 g (n = 230) (P < 0.001). Female birthweight also decreased from 3148 ± 378 (n = 377) to 3063 ± 343 g (n = 212) (P < 0.01). Gestational age was shorter (40.1 ± 1.1 to 39.6 ± 1.1 weeks, P < 0.001). Pre-pregnancy maternal BMI did not change (20.7 ± 2.6, n = 750 to 20.5 ± 2.7, n = 442). Weight gain during pregnancy decreased (11.3 ± 3.8 to 10.8 ± 3.4 kg, P < 0.05) and was positively correlated with birthweight (P < 0.001). On multiple regression analysis, time was inversely correlated with birthweight, while it was positively correlated with pre-pregnancy BMI, weight gain during pregnancy, and gestational age. CONCLUSIONS: In addition to maternal slenderness, increasing lower weight gain during pregnancy and medical intervention by physicians may in part contribute to the decrease in full-term birthweight over the past 2 decades.

Fourteen-year-old girl with recurrent luteal-phase-dependent episodes of high fever.

Recurrent episodes of high fever during the luteal phase are exceedingly rare. A 14-year-old girl with a 1-year history of recurrent febrile episodes associated with the menstrual cycle was referred to our department. Febrile episode (39-41°C) occurred for 10 to 12 days concomitantly with the luteal phase of each ovulatory cycle. To suppress the ovulatory cycle, gonadotropin releasing hormone agonist was administered for 5 months, and then febrile attack disappeared (34.5-36.2°C). Before and during gonadotropin releasing hormone agonist therapy, serum levels of inflammatory cytokines (interleukin [IL]-1ß, IL-2, IL-6, IL-8, IL-10, and tumor necrosis factor-α) were unchanged and all of these were within the normal ranges. In our case, luteal-phase-dependent febrile episodes may not be attributable to the intervention of inflammatory cytokines as major fever mediators.

Relative contribution of weight-bearing and non-weight-bearing effect of adipose tissue to bone mineral density in postmenopausal women.

AIM: To investigate the relative contributions of weight-bearing and non-weight-bearing effects of adipose tissue to bone mineral density (BMD) in postmenopausal women. MATERIAL AND METHODS: The subjects were 228 postmenopausal women aged 50-75 years. Age, years since menopause (YSM), height, body weight, and body mass index were recorded. Trunk fat mass, body fat mass, bilateral leg BMD and lean (muscle) mass were measured by whole body scanning with dual-energy X-ray absorptiometry. The relationships of BMD to trunk and body fat mass were investigated using uni- and multivariable analyses. RESULTS: The amount of trunk fat mass and body fat mass were 8.7 ± 3.6 kg and 19.0 ± 5.9 kg, respectively. On Pearson's correlation test, right leg BMD was positively correlated with trunk fat mass (r=0.268, P<0.001) and body fat mass (0.299, P<0.001). On multiple linear regression analysis, trunk fat mass (t-value = 3.500, P<0.001), age (-2.431, P<0.05), and YSM (-2.564, P<0.01) were independent significant predictors of right leg BMD. However, body fat mass was not a predictor of BMD (-0.465, P=0.642). These relationships remained significant after further adjusting for right leg muscle mass. CONCLUSION: Trunk fat mass rather than body fat mass is a significant predictor of leg BMD at the most weight-bearing site, despite being less than half the amount of body fat mass. Thus, adipose tissue contributes more to BMD through non-weight-bearing effect rather than weight-bearing effect.

Difference in non-weight-bearing effects on bone mineral density between trunk and peripheral fat mass in women with polycystic ovary syndrome.

AIM: To investigate the difference in non-weight-bearing effects on bone mineral density (BMD) between trunk and peripheral fat mass in women with polycystic ovary syndrome (PCOS). METHODS: Subjects were 123 amenorrheic PCOS women with right side dominance. Age, height, body weight, and body mass index were recorded. Trunk, peripheral (extremities), trunk-leg fat ratio as an index of body fat distribution, left arm (non-weight-bearing site) lean mass and BMD were measured by dual-energy X-ray absorptiometry. Serum testosterone and estradiol levels were measured. Relationships of BMD with trunk, peripheral fat mass, and sex hormones levels were investigated. RESULTS: Trunk fat mass amount was 9.8 + or - 6.7 kg and was lower than the peripheral fat mass amount (12.2 + or - 4.4 kg, P < 0.01). On Pearson's correlation test, trunk fat mass and left arm lean mass were positively correlated with arm BMD (r = 0.359, P < 0.001 and r = 0.501, P < 0.0001, respectively), while peripheral fat mass and serum testosterone levels were not correlated with BMD (r = 0.083 and 0.114, respectively, NS). On multiple regression analysis, trunk fat mass was positively correlated with BMD (t-value = 3.465; P < 0.001), independent of age and height. However, this relationship disappeared after additionally adjusting for left arm lean mass. CONCLUSION: Trunk fat mass, despite the smaller amount, is more associated with arm BMD than peripheral fat mass is through its non-weight-bearing effects.

[Case of anti-glomerular basement membrane antibody nephritis in a patient who was able to withdraw from dialysis and gave birth twice but underwent living renal transplantation due to progression to end-stage renal failure 15 years after onset].

A 34-year-old woman with suspected rapidly progressive glomerulonephritis had been admitted to our hospital in March 1993 at the age of 19 years. Renal biopsy revealed cellular crescent formation in 24 of 26 glomeruli. Serum examination was positive for anti-glomerular basement membrane (GBM) antibody, while pulmonary hemorrhage was absent. Based on these findings, she was diagnosed with anti-GBM antibody nephritis, and treated with corticosteroid pulse therapy and double filtration plasmapheresis (DFPP) in addition to hemodialysis (HD). HD was withdrawn within 2 months. Wishing to have a baby, she had delivery in 1997 and 2000. Subsequently, her renal function gradually decreased, and she underwent an ABO-incompatible living-donor kidney transplant, with her husband as the donor, in March 2008. She has been making good progress after transplantation. Anti-GBM antibody nephritis has a poor prognosis, but renal function was maintained for 15 years in this patient, who responded well to the initial treatment. The underlying disease rarely recurs if transplantation is performed after the patient has become negative for anti-GBM antibody, anti-GBM antibody nephritis therefore seems to be a good indication for treating patients with renal transplantation.

Non-weight-bearing effect of trunk and peripheral fat mass on bone mineral density in pre- and post-menopausal women.

OBJECTIVE: To investigate the non-weight-bearing effect of trunk fat mass (composed of visceral and subcutaneous fat mass) and peripheral fat mass (subcutaneous fat mass alone) on bone mineral density (BMD) in pre- and post-menopausal women. METHODS: The subjects were 412 pre-menopausal women, 20-50 years of age and 228 post-menopausal women, 50-75 years of age. Age, years since menopause (YSM), height, body weight, and body mass index were recorded. Trunk, peripheral (extremities), left arm (non-weight-bearing site), lean mass, and BMD were measured by dual-energy X-ray absorptiometry. RESULTS: In pre-menopausal women, the amount of trunk fat mass was 6.8+/-4.1 kg, which was significantly lower than the amount of peripheral fat mass (11.6+/-3.8 kg, p < 0.001). Although trunk fat mass was positively correlated with arm BMD on Pearson's correlation test, arm lean mass was the only significant predictor of BMD on multiple regression analysis. In post-menopausal women, the amount of trunk fat mass (8.7+/-3.6 kg) was also significantly lower than the peripheral fat mass (10.3+/-3.4 kg, p < 0.001). On multiple regression analysis, however, trunk fat mass, but not arm lean mass, was the significant predictor of BMD. In both groups, peripheral fat mass was not correlated with left arm BMD. CONCLUSION: The effect of adipocyte-derived biochemical factors on BMD may differ with menopausal status and the sites of adipocyte deposition.

Chylous vaginal discharge in a patient with lymphangioleiomyomatosis.

We present the case of a patient having chylous vaginal discharge, which may be a symptom of the secondary to an established diagnosis of the systemic disorder, lymphangioleiomyomatosis, a disease predominantly affecting women during their reproductive years. Lymphangioleiomyomatosis causes dyspnea, pneumothorax, chylous pleural effusions, and eventually respiratory failure.

Retroperitoneal uterine leiomyoma occurring 5 years after hysterectomy for fibroids.

We encountered a 49-year-old, multiparous female with a very rare isolated retroperitoneal uterine leiomyoma measuring 72 x 43 mm in diameter occurring 5 years after hysterectomy for fibroids. The case was preliminarily diagnosed as right ovarian cancer or fibroma. An edematous, isolated solid tumor in the right retroperitoneal cavity was surgically resected. Pathological findings demonstrated uterine leiomyoma.

Aromatase inhibitor-induced bone mineral loss and its prevention by bisphosphonate administration in postmenopausal breast cancer patients.

AIM: To investigate aromatase inhibitor-induced bone mineral loss and its prevention by bisphosphonate administration in postmenopausal breast cancer patients. METHODS: Subjects were 17 postmenopausal breast cancer patients (mean age, 63.3 +/- 9.9 years) receiving non-steroidal aromatase inhibitor (AI; anastrozole, 1 mg daily) only and 10 such patients (mean age, 65.0 +/- 5.1 years) receiving AI + bisphosphonate (risedronate sodium, 2.5 mg daily) for 6 months. All of the subjects had undergone surgical resection and had positive estrogen receptor tumor status. Age, age at menopause, years since menopause, height, weight, and body mass index (Wt/Ht(2)) were recorded. Lumbar spine (L2-4) bone mineral density (BMD), T-, and Z-scores were assessed on dual-energy X-ray absorptiometry before and after therapy. RESULTS: In the AI-only group BMD, T-, and Z-scores significantly decreased from the baseline during the 6-month therapy period (P < 0.05). Mean decreases in L2-4 BMD and Z-score were 2.5% and 3.0%, respectively. In the AI + bisphosphonate group, however, BMD, T-, and Z-scores significantly increased from the baseline values (P < 0.01). Mean increases in L2-4 BMD and Z-score were 4.5% and 3.3%, respectively. CONCLUSION: AI carries a potential risk of bone mineral loss despite the short therapy duration. Bisphosphonate has a preventive effect on this loss.

Perforated colon volvulus coiling around the uterus in a pregnant woman with a history of severe constipation.

A case of intestinal volvulus in both the sigmoid colon and cecum in a pregnant woman suffering from severe constipation is reported herein. The fetus was dead and the mother developed acute shock. The twisted sigmoid colon coiled twice around the uterus and was perforated. The mother died 10 h after the onset of severe abdominal pain. Volvulus should be considered when examining severe abdominal pain in a pregnant woman with a history of severe constipation. Early suspicion together with prompt intervention will minimize maternal and fetal morbidity and mortality of this rare complication of pregnancy.

Mechanical ileus in a pregnant woman at term pregnancy accompanied by labor pains.

Intestinal obstruction in pregnancy is a rare, but serious complication of pregnancy with significant maternal and fetal mortality. We herein report a case of intestinal strangulation in a pregnant woman with a history of pelvic surgery due to an ectopic pregnancy. Epigastric pain occurred at term pregnancy with concomitant onset of labor pains. The epigastric pain disappeared transiently, and she gave a birth to a healthy child. However, the pain appeared again after the vaginal delivery. She immediately underwent ileo-ileostomy with a diagnosis of mechanical ileus, and the postoperative course was uneventful. Mechanical ileus should be considered when examining epigastric pain in a pregnant woman with a history of abdominal or pelvic surgery even after the onset of labor pains.

Isolated adenomyotic cyst associated with severe dysmenorrhea.

A case of a 23-year-old, nulliparous female with a very rare isolated adenomyotic cyst inducing severe dysmenorrhea was seen. Transvaginal ultrasonographic tomography and magnetic resonance imaging (MRI) showed a 3 x 3-cm cystic mass within the left anterior wall of the uterine corpus. The cystic space was filled with hyperintense fluid on T1-weighted images, which was surrounded by hypointense tissue beside the right uterine corpus on T2-weighted images. The case was preliminarily diagnosed using MRI as having cavitated rudimentary uterine horn. However, hysterosalpingography excluded the possibility of uterine anomaly. A hemorrhagic adenomyotic cyst measuring 3 cm within the left anterior wall of the uterine corpus was surgically removed. There was no evidence of diffuse adenomyosis uteri. Dysmenorrhea completely disappeared postoperatively.

Difference in segmental lean and fat mass components between pre- and postmenopausal women.

OBJECTIVE: To investigate the differences in segmental body composition (lean and fat mass components) between pre- and postmenopausal women. DESIGN: Participants were 413 premenopausal women aged 20 to 53 years old and 229 postmenopausal women aged 50 to 75 years old with right-side dominance. Age, height, weight, body mass index, age at menopause, and years since menopause were recorded. The percentages of fat mass in the arms, trunk, legs, and total body were measured by dual-energy x-ray absorptiometry. The ratio of trunk to leg fat mass (trunk-leg fat mass ratio) was also measured by dual-energy x-ray absorptiometry. RESULTS: The percentage of trunk fat mass and the trunk-leg fat mass ratio were significantly higher in postmenopausal women, but the percentages of leg fat mass did not differ. In the two groups, percentage of trunk fat mass and trunk-leg fat mass ratio were similarly and positively correlated with age. However, percentage of leg fat mass did not correlate with age. The percentage of fat mass at each segmental site and the trunk-leg fat mass ratio did not differ between premenopausal women aged 50 to 53 years old (n=52) and age-matched postmenopausal women (n=43, years since menopause=2.8+/-1.8). CONCLUSIONS: Aging rather than menopause contributes to the increase in the percentage of trunk fat mass. However, the percentage of leg fat mass does not change with aging. Upper body fat distribution in postmenopausal women may be more attributable to aging than to menopause.

Bone mineral density in breast cancer patients with positive estrogen receptor tumor status.

AIM: The aim was to investigate bone mineral density (BMD) in breast cancer patients with positive estrogen receptor (ER) tumor status. METHODS: The participants were 110 postmenopausal breast cancer patients with positive estrogen receptor (ER+) tumor status. Two hundred and sixty-one age-matched, healthy postmenopausal women, all of whom were selected from our pooled data, served as controls. Age, age at menopause, years since menopause (YSM), height, weight, and body mass index (BMI, wt/ht(2)) were recorded. Lumbar spine (L2-4) BMD and Z-score were assessed by dual-energy X-ray absorptiometry. RESULTS: Bone mineral density in breast cancer patients was significantly higher than that in controls (0.89+/-0.12 g/cm(2) versus 0.84+/-0.16 g/cm(2), P<0.01). The Z-score in breast cancer patients was also higher than that in controls (110+/-13.6% versus 100+/-9.8%, P<0.001). Higher BMD and Z-score in breast cancer patients remained significant after adjusting for age, YSM, and BMI (P<0.05). CONCLUSIONS: Postmenopausal breast cancer patients with positive ER tumor status have higher BMD. Positive ER tumor status may be associated with higher cumulative exposure to estrogen.

Severe leg compartment syndrome associated with dorsal lithotomy position during radical hysterectomy.

We encountered a female patient with left-leg compartment syndrome; a devastating complication, probably associated with prolonged dorsal lithotomy position during radical hysterectomy using intermittent pneumatic compression. This patient was intensively treated and fortunately recovered. However, leg compartment syndrome is poorly understood by gynecologists. We must always consider the potential risk of this life-threatening complication when patients are placed in the dorsal lithotomy position for a prolonged period during extended surgery using intermittent external compression.

Precedence of the shift of body-fat distribution over the change in body composition after menopause.

AIM: This study investigated the sequence of certain phenomena after menopause: decrease in bone mineral density (BMD), change in body composition (lean and fat components), and the shift toward upper body fat distribution. METHODS: Subjects were 188 postmenopausal women aged 50-65 years old. They were divided into four subgroups based on 4-year increments in age. Regularly menstruating women (n = 51) aged 50-53 years old served as controls. Age, height, weight, and years since menopause were recorded. Body fat mass, percentage of body fat (%fat), lean body mass (LBM), lumbar spine (L2-4), total body BMD, and the trunk-leg fat mass ratio were measured by dual-energy X-ray absorptiometry. RESULTS: In postmenopausal women (n = 42) aged 50-53 years, BMD was lower compared to age-matched controls (P < 0.05), while other variables did not differ. Trunk-leg fat mass ratio in women aged 54-57 years or more was significantly higher than that in control. LBM was significantly lower while percentage fat was significantly higher in women aged 58-61 years old or more. CONCLUSION: An initial event during the menopausal process is BMD loss, which is followed by body fat distribution shift, then LBM loss and reciprocal increase in body fat mass.

Preoperative plasma osteopontin level as a biomarker complementary to carbohydrate antigen 125 in predicting ovarian cancer.

AIM: New biomarkers other than carbohydrate antigen (CA) 125 are needed for the detection of ovarian cancer. Osteopontin (OPN) is one of the candidates identified by high-throughput complementary DNA microarray techniques. We evaluated the preoperative plasma OPN level as a diagnostic biomarker for ovarian cancer in comparison with CA125. METHODS: Preoperative plasma OPN and CA125 levels were measured and compared in 32 patients with ovarian cancer, 34 patients with benign ovarian tumor, 30 patients with other gynecologic cancers and 31 healthy women. Preoperative plasma OPN levels were also assessed according to tumor stage, the volume of ascites and histological types. The sensitivity and specificity for predicting ovarian cancer was compared between OPN and CA125. RESULTS: Preoperative plasma OPN levels were significantly higher in patients with ovarian cancer than in those with benign ovarian tumor, in other gynecologic patients or in healthy women. Stage IV ovarian cancer patients and ovarian cancer patients with ascites had higher plasma OPN levels than those without ascites and in a lower stage. There was no relation between OPN and the histological type. The sensitivity of preoperative plasma OPN in detecting ovarian cancer was 81.3% and almost reached that of CA125. The specificity was moderate. Sensitivity increased to 93.8% with the combination of CA125, compared to 84.4% with CA125 alone. CONCLUSION: Preoperative OPN is a useful biomarker for predicting ovarian cancer. It is especially useful when used complementary to CA125. Larger studies of patients with ovarian cancer showing a low CA125 level or in early stages of ovarian cancer are needed.

Difference in the relative contribution of lean and fat mass components to bone mineral density with generation.

AIM: To investigate whether the relative contribution of body composition (lean and fat mass component) to bone mineral density (BMD) differs depending on generation or menopause. METHODS: Subjects were 302 premenopausal women aged 30-49 years old and 197 postmenopausal women aged 50-69 years old. They were classified into four subgroups with 10-year increments. Age, height, weight and years since menopause (YSM) were recorded. Lumbar spine (L2-4), total body BMD, body fat mass, lean body mass (LBM), and the percentage of body fat (%fat) were measured using dual-energy X-ray absorptiometry. The correlation of body composition with BMD was investigated. RESULTS: The mean age at menopause was 50.2 +/- 4.1 years old. On Pearson's correlation test, LBM was positively correlated with BMD of the two sites in all groups. In a group aged 60-69 years, both the %fat and body fat mass were correlated with BMD. On multiple regression analysis, LBM was the principal BMD determinant in women aged less than 60 years, while body fat mass and percentage fat were the principal BMD determinants in women aged 60-69 years. Mean %fat in the group aged 60-69 years was 35.5 +/- 7.3%, which was significantly higher than that in the group aged 50-59 years (33.0 +/- 6.7%, P < 0.05). CONCLUSION: Although LBM still influences BMD up to 10 years after menopause, the body fat mass initially influences BMD after 60 years of age. These difference may be attributable to certain aging-related factor(s).

Metastasis of uterine leiomyosarcoma to the pancreas.

BACKGROUND: Metastasis of uterine leiomyosarcoma to the pancreas is extremely rare. CASE: A 46-year-old woman presented with hypermenorrhea and dysmenorrhea and underwent surgery. The histologic and immunohistochemical diagnosis was uterine leiomyosarcoma stage I with no metastasis to the ovaries or the pelvic lymph nodes. The mitotic count was very high. Thereafter, recurrences in the lung and subsequently in the pancreas were detected. The lesions in the lung and pancreas were resected and diagnosed as metastases of uterine leiomyosarcoma based on histology and immunohistochemistry. CONCLUSION: We report an extraordinarily rare case of uterine leiomyosarcoma with metastasis to the pancreas following initial metastasis to the lung, both of which were diagnosed using histology and immunohistochemistry of the specimens obtained at surgery.