Two consecutive successful live birth in woman with 17α hydroxylase deficiency by frozen-thaw embryo transfer under hormone replacement endometrium preparation.

17α-Hydroxylase deficiency is rare autosomal recessive disorder that manifested by hypertension, hypokalemia, delayed sexual development, primary amenorrhea and infertility. The information regarding infertility care and conception in women with this disorder are extremely limited. We report a 24-year-old Japanese woman with primary amenorrhea who was diagnosed as partial 17α-hydroxylase deficiency caused by homozygous 3 bp deletion in exon 1 of 17α-hydroxylase gene. In vitro fertilization with controlled ovarian stimulation was carried out and all viable embryo were frozen. During ovarian stimulation, serum progesterone levels were markedly elevated, and endometrial growth was impaired. Utilizing frozen-thaw embryo transfer under hormonal replacement (glucocorticoid, estradiol and progesterone), she had successfully given two consecutive live birth. Women with 17α-hydroxylase deficiency with residual ovarian reserve can afford reproductive success by appropriate diagnosis and treatment by assisted reproductive technology.

Association between ovarian endometrioma and ovarian reserve.

Endometrioma is one of the main pathologies of endometriosis, though its pathogenesis still remains enigmatic. Ovarian reserve is defined as the number and quality of the follicles left in the ovary at any given time. The cause of infertility in women with endometriosis is multifactorial. Diminished ovarian reserve is major concern in women with endometriosis-associated infertility. Cystectomy for endometriomas could negatively impact on post-operative ovarian reserve. Some women had surgery for endometriomas suffer from poor ovarian response, which directly affects treatment results. In addition, endometriomas themselves may be a cause of diminished ovarian reserve. Destruction of normal histological structure in ovarian cortex may affect dormancy of primordial follicles. Therefore, determination of ovarian reserve may serve as an important role in the management of reproductive health of women with endometriosis. Although the knowledge on the physiology of follicular development and mechanism of maintenance of ovarian reserve are rapidly accumulating, results obtained by ovarian reserve testing after surgery should be carefully evaluated according to the time-points and selected test. Further investigation on this issue is warranted.

Three-dimensional saline-infusion sonohysterography is useful for the identification of endometrial polyp.

AIM: To determine whether three-dimensional saline infusion sonohysterography (3D-SISH) is useful for the detection of endometrial polyps and whether it can accurately identify the site of attachment within the uterine cavity. METHODS: Sixteen women who underwent preoperative magnetic resonance imaging (MRI), 3D-SISH and subsequent hysteroscopic resection or total hysterectomy and who were surgically diagnosed with endometrial polyps were enrolled. The rate of polyp detection and the accuracy of attachment site within the uterine cavity for each test were examined. RESULTS: Endometrial polyps could only be identified in 37.5% of women using MRI, but could be identified in all women using 3D-SISH. The accuracy rate of the attachment site of endometrial polyps was 87.5% on 3D-SISH and 18.8% (in all patients) or 50.0% (in polyp-detected patients) on MRI, indicating a higher accuracy rate using 3D-SISH. CONCLUSION: We demonstrated that 3D-SISH is useful for the detection and localization of endometrial polyps and is likely to become the examination method of first choice for the identification of endometrial polyps.

17ß-estradiol and lipopolysaccharide additively promote pelvic inflammation and growth of endometriosis.

Endometriosis is a multifactorial disease mostly affecting women of reproductive age. An additive effect between inflammation and stress reaction on the growth of endometriosis has been demonstrated. Here we investigated the combined effect between 17ß-estradiol (E2) and lipopolysaccharide (LPS) on pelvic inflammation and growth of endometriotic cells. Peritoneal fluid was collected from 46 women with endometriosis and 30 control women during laparoscopy. Peritoneal macrophages (Mφ) and stromal cells from eutopic/ectopic endometrial stromal cells (ESCs) were isolated from 10 women each with and without endometriosis in primary culture. Changes in cytokine secretion (interleukin 6 [IL-6] and tumor necrosis factor α [TNF-α]) by Mφ and proliferation of ESCs in response to single and combined treatment with E2 and LPS were measured by enzyme-linked immunosorbent assay and by bromodeoxyuridine incorporation assay, respectively. A significantly increased secretion of IL-6 and TNF-α in Mφ culture media was found in response to E2 (10(-8) mol/L) compared to nontreated Mφ. This effect of E2 was abrogated after pretreatment of cells with ICI 182720 (10(-6) mol/L; an estrogen receptor [ER] antagonist). Combined treatment with E2 and LPS (10 ng/mL) additively promoted IL-6 and TNF-α secretion by peritoneal Mφ and growth of eutopic/ectopic ESCs. The additive effects of E2 + LPS on cytokine secretion and growth of ESCs were effectively suppressed after combined blocking of ER and Toll-like receptor 4. An additive effect was observed between E2 and LPS on promoting proinflammatory response in pelvis and growth of endometriosis.

Evaluation of obstetrical factors related to Sheehan syndrome.

AIM: To evaluate factors related to the occurrence of Sheehan syndrome. METHODS: The obstetrical disseminated intravascular coagulation score, total volume of hemorrhage, shock index, level of consciousness at the time of shock occurrence and pituitary magnetic resonance imaging findings were evaluated in nine women who showed massive hemorrhage during delivery. These clinical outcomes were analyzed in all these patients who were prospectively followed-up to identify any possible occurrence of Sheehan syndrome. RESULTS: Compared to six women with non-Sheehan syndrome, three women who were diagnosed with Sheehan syndrome showed significant elevation of the obstetrical disseminated intravascular coagulation score, decrease in the level of consciousness during shock and remarkable pituitary gland atrophic change with an empty sella turcica detected by pituitary magnetic resonance imaging. The volume of hemorrhage during delivery and shock index were not significantly different between these two groups of women. CONCLUSION: Careful attention and follow-up should be paid to women with post-partum massive hemorrhage for early detection and management of women with Sheehan syndrome.

Additive effects of inflammation and stress reaction on Toll-like receptor 4-mediated growth of endometriotic stromal cells.

STUDY QUESTION: Is there any combined effect between inflammation and stress reaction on Toll-like receptor 4 (TLR4)-mediated growth of endometriotic cells? SUMMARY ANSWER: A combined effect of local inflammation and stress reaction in the pelvic environment may be involved in TLR4-mediated growth of endometriotic stromal cells. WHAT IS KNOWN ALREADY: In endometriosis, higher endotoxin levels in the menstrual fluid (MF) and peritoneal fluid (PF) and higher tissue concentrations of human heat shock protein 70 (HSP70) in the eutopic and ectopic endometria promote TLR4-mediated growth of endometriotic cells. STUDY DESIGN, SIZE AND DURATION: This is a case-controlled research study with prospective collection and retrospective evaluation of sera, MF, PF and endometrial tissues from 43 women with and 20 women without endometriosis. PARTICIPANTS/MATERIALS, SETTING, METHODS: PF was collected from 43 women with endometriosis and 20 control women during laparoscopy. Sera and endometrial biopsy specimens were collected from a proportion of these women. MF was collected from a separate population of 20 women with endometriosis and 15 control women. HSP70 concentrations in sera, MF, PF and in culture media were measured by ELISA. Gene expression of HSP70 by endometrial cells in response to lipopolysaccharide (LPS) was examined by qRT-PCR. The individual and combined effects of LPS and HSP70 on the secretion of interleukin-6 (IL-6) and tumor necrosis factor α (TNFα) by PF-derived macrophages (M[Symbol: see text]) were examined by ELISA, while their effects on endometrial cell proliferation were examined by bromodeoxyuridine and [(3)H]-thymidine incorporation assay. MAIN RESULTS AND THE ROLE OF CHANCE: Concentrations of HSP70 were maximal in MF, intermediate in PF and the lowest in sera. In MF and PF, HSP70 levels were higher in women with endometriosis than in controls. LPS stimulated gene expression and secretion of HSP70 by eutopic endometrial stromal cells (ESCs) and this effect was abrogated after pretreatment of cells with an anti-TLR4 antibody. This effect was significantly higher for ESCs derived from women with endometriosis than for ESCs from control women. Exogenous treatment with either HSP70 or LPS significantly stimulated the production of IL-6 and TNFα by M[Symbol: see text] and promoted the proliferation of ESCs, and a significant additive effect between LPS and HSP70 was observed. While individual treatment with either polymyxin B, an LPS antagonist, or anti-HSP70 antibody was unable to suppress the combined effects of LPS and HSP70 on cytokine secretion or ESC proliferation, pretreatment of cells with the anti-TLR4 antibody was able to significantly suppress their combined effects. LIMITATIONS, REASONS FOR CAUTIONS: Further studies are needed to examine the mutual role between other secondary inflammatory mediators and endogenous stress proteins in promoting pelvic inflammation and growth of endometriotic stromal cells. WIDER IMPLICATIONS OF THE FINDINGS: Our findings suggest that endotoxin and HSP70 are mutually involved in a stress reaction and in inflammation. A combined effect between local inflammation and a stress reaction in pelvic environment may be involved in TLR4-mediated growth of endometriotic cells. Since endometriosis is a multi-factorial disease, it is difficult to explain uniformly its growth regulation by a single factor. Our findings may provide some new insights in understanding the physiopathology or pathogenesis of endometriosis and may hold new therapeutic potential. STUDY FUNDING/COMPETING INTEREST(S): This work was supported by Grants-in-Aid for Scientific Research (grant no. 16591671 and 18591837) from the Ministry of Education, Sports, Culture, Science and Technology of Japan (to K.N.K.). There is no conflict of interest related to this study. TRIAL REGISTRATION NUMBER: Not applicable.

Developing the clinical abilities of care workers in Japan.

The increasing number of frail older people being admitted to Japan's care homes makes it difficult for healthcare professionals alone to attend to all their needs. By law, care workers are not allowed to carry out any clinical procedures but, in response to the situation, the Japanese Ministry of Health, Labour and Welfare has allowed this group of workers to carry out limited clinical activities, such as oral suctioning and percutaneous endoscopic gastrostomy tube feeding at one type of Japanese care home, welfare care homes. Care workers must be supervised by healthcare professionals and follow guidelines. The ministry has further decided to introduce a certification system, so that from 2015 certified care workers will be able to engage lawfully in these activities.

Changes in serum anti-Müllerian hormone levels may predict damage to residual normal ovarian tissue after laparoscopic surgery for women with ovarian endometrioma.

We measured serum anti-Müllerian hormone levels before and after surgery in women undergoing unilateral and monolocular cystectomy for benign ovarian diseases. Comparing to control benign cysts, we found a significant decline in serum anti-Müllerian hormone levels with consequent depletion of follicles in tissue specimens after surgery for women with ovarian endometrioma.

Placental multiple chorionic cysts in maternal scleroderma.

We present a case of a pregnant woman with scleroderma (Ssc) whose placenta showed multiple chorionic cysts and severe fibrotic changes and large infarcted areas at the maternal side. Fetal growth was appropriate for gestational age and amniotic fluid volume was normal, but fetal tachycardia, loss of variability, and late deceleration were detected by non-stress test at 29 weeks of gestation. She was diagnosed as having non-reassuring fetal status and delivered a female baby who weighed 1092 g (Apgar score 6/9) by Caesarean section. Placental surface cysts are rare findings and their effect on pregnancy is poorly understood, but an association with intrauterine growth restriction (IUGR) has been reported. This is the first report of a pregnant woman with scleroderma showing multiple placental cysts.

Oncocytic carcinoma of the nasal cavity with widespread lymph node metastases.

The first case of oncocytic carcinoma which arose from the inferior turbinate of the nasal cavity with orbital invasion through the nasolacrimal canal and widespread lymph node metastases in the neck and face is reported here. The tumor metastasized rapidly, spread widely to the whole neck and face, and produced tumor emboli in the lymphatics and extranodal extension by rupture of the lymph node capsule in the absence of clinically palpable regional lymph node enlargement. We think the oncocytic carcinoma of the nasal cavity may be a high-grade malignancy tumor.

Solitary fibrous tumor arising in the sphenoethmoidal recess: a case report and review of the literature.

Recently, solitary fibrous tumors (SFTs) have been reported in the head and neck area, such as the nasal cavity, thyroid, salivary gland, etc. We present a rare case of SFT which arose from the sphenoethmoidal recess of the nasal cavity, penetrating into the sphenoid sinus, and which showed different intensities on magnetic resonance imaging (MRI) according to the occupied locations. T2 weighted magnetic resonance (MR) images showed low intensity in the nasal cavity, and iso-intensity in the sphenoid sinus. Enhancement with gadolinium contrast on T1-weighted images was more remarkable in the sphenoid sinus than in the nasal cavity. While the tumor in the nasal cavity showed abundant collagen and high cellularity in microscopic examination, numerous small vessels and dilated vascular spaces were remarkable in the tumor of the sphenoid sinus. MRI findings corresponded to pathological findings. We review SFTs in the head and neck area in the English literature.

Increases in serum nitrite and nitrate of a few-fold adversely affect the outcome of pregnancy in rats.

The objective of this study was to evaluate serum nitrite and nitrate (nitrite/nitrate) concentrations that affect adversely pregnancy outcome. Pregnant rats, from day 2 to day 8 of pregnancy, were daily given subcutaneously several doses (5, 10, and 30 mg/rat) of diethylenetriamine-nitric oxide (DETA/NO). Serum nitrite/nitrate concentrations were measured using an HPLC system. Serum nitrite/nitrate concentrations increased dose-dependently with DETA/NO. Effects of DETA/NO on pregnancy outcome were assessed on day 14 of pregnancy. In rats given 5 mg DETA/NO, there was a significant increase in serum nitrite/nitrate concentrations (49.2 vs 24.6 micromol/l, P<0.001), and both placental weight and fetal weight decreased compared to control rats. Macroscopic bleeding in placenta was frequently observed in rats given DETA/NO. We further studied effects of DETA/NO on cultured trophoblastic BeWo cells. DETA/NO added to the culture medium increased nitrite/nitrate concentrations in the medium in a dose-dependent manner. Nitrite/nitrate concentrations in the medium over four times the concentration of control decreased progesterone in the medium at 24 h after the application of DETA/NO. The hormonal secretion was not affected by DETA only. This study shows for the first time nitrite/nitrate concentrations affecting adversely pregnancy outcome and function of the trophoblastic cells.