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ABSTRACT: Sacrococcygeal teratoma (SCT) is a rare congenital disease and prognostic factors have not been entirely established. We report two cases of fetal SCT with different clinical courses. Case 1 was a cystic, slow growing tumor with mild vascularity. The tumor was removed one week after delivery at 35 weeks, and there was no recurrence at 1.5-year follow-up. Case 2 was a solid, rapid growing tumor with rich vascularity. Cesarean section was performed due to severe fetal hydrops and mirror syndrome in the mother at 27 weeks. The tumor had ruptured and was removed soon after delivery to control bleeding, but the baby died the next day. Our cases suggest that solid component and rich vascularity might correlate with poor prognosis.
Congenital Anomalies 06/2013; 53(2):92-4.
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ABSTRACT: OBJECTIVE: To compare follicular fluid and serum concentrations of anti-Mullerian hormone (AMH) as predictors of the outcome of assisted reproduction. STUDY DESIGN: This observational study enrolled 58 women who were undergoing IVF or ICSI treatment with the long stimulation protocol. Patients diagnosed as having PCOS were excluded. Serum and follicular fluid AMH levels were assessed as predictors of clinical pregnancy. RESULTS: Both the serum and follicular fluid AMH levels were higher in the clinical pregnancy group than in the failed group. A significant correlation was found between the serum and follicular fluid AMH levels, but a discrepancy was observed in some patients with elevated AMH levels in the follicular fluid or serum alone. Assisted reproductive treatment resulted in clinical pregnancy in all of the patients with elevated AMH levels in the follicular fluid (>40pM) or in the serum (>10pM). The ROC-AUC for the combination of follicular and serum AMH was 0.772, which was relatively higher than that for either the serum AMH (AUC: 0.691) or follicular fluid AMH (AUC: 0.688) alone. CONCLUSION(S): Elevated AMH levels in either the serum or follicular fluid appeared to be predictive of clinical pregnancy, even if AMH levels in other fluids were low. This is a pilot study with preliminary data that need further confirmation.
European journal of obstetrics, gynecology, and reproductive biology 04/2013; · 1.97 Impact Factor
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ABSTRACT: Omphalocele-exstrophy of the bladder (cloaca)-imperforate anus-spinal defects (OEIS) complex describes a rare grouping of more commonly occurring component malformations. We report two cases of OEIS complex diagnosed prenatally by ultrasound and magnetic resonance imaging (MRI). In both cases, OEIS complex was suspected by conventional sonography in the second trimester, and fetal MRI was performed at 27 and 32 weeks of gestation. Conventional sonography revealed low abdominal wall mass, spina bifida, absent bladder and ambiguous genitalia, but those findings were inconclusive. Using fetal MRI, we were able to detect omphalocele, imfraumbilical mass connected to gut tract, absent bladder, ambiguous external genitalia and spinal defect. Our findings suggest that fetal MRI is a useful tool for prenatal diagnosis of OEIS complex. MRI helps prenatal counseling and planning of postnatal early treatment strategy.
Congenital Anomalies 09/2012; 52(3):179-81.
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ABSTRACT: Aims: The aim of this study was to examine the influence of recurrent spontaneous abortion (RSA) on marital relationships, and the association between past/present illness and RSA. Material and Methods: A total of 2733 Japanese women who underwent a medical examination responded to the questionnaire. Results: The frequency of recurrent miscarriage and two or more consecutive RSA were 0.88% and 4.2%, respectively. Women with a history of miscarriages (hazard ratio: 1.596) and RSA (hazard ratio: 3.103) were at a higher risk of their relationships ending as compared with the women without a history of miscarriage. Existence of a relation was seen between a history of RSA and the occurrence of gastric ulcer, gastritis, fatty liver, and atopic dermatitis. Overall, 89.5% of the women with RSA experienced cumulative live births. Conclusions: Miscarriage was found to be a severe life event with an influence on marital relationships, and to be associated with an elevated risk of gastric disease or atopic dermatitis.
Journal of Obstetrics and Gynaecology Research 08/2012; · 0.94 Impact Factor
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ABSTRACT: We previously found that a normal karyotype in a previous miscarriage is a predictor of subsequent miscarriage. However, the prevalence of recurrent miscarriage caused by an abnormal embryonic karyotype has not yet been reported, since embryonic karyotype is not typically analyzed during conventional examinations.
A total of 482 patients who underwent both embryonic karyotype determination and conventional examinations for recurrent miscarriage were enrolled in this study. The distribution of the causes and the live birth rate for each cause were examined.
The total percentage of subjects in whom conventional causes of recurrent miscarriage could be detected was 29.5%. The prevalence of the abnormal embryonic karyotype was 41.1% in the subjects in whom no conventional causes of miscarriage could be identified. The prevalence of recurrent miscarriage of truly unexplained cause, that is, of subjects without conventional causes in whom the embryonic karyotype was ascertained to be normal, was 24.5%. Among the patients in whom the first determination revealed an abnormal embryonic karyotype, 76.2% (32/42) showed an abnormal embryonic karyotype in the repeat determination as well. The cumulative live birth rate (71.9%) in women with recurrent miscarriages caused by the abnormal embryonic karyotype was significantly higher than that (44.7%) in women with recurrent miscarriages associated with the embryonal euploidy.
An abnormal embryonic karyotype was found to represent the commonest cause of recurrent miscarriage, and the percentage of cases with recurrent miscarriage of truly unexplained cause was limited to 24.5%.The two groups should be distinguished for both clinical and research purposes.
Human Reproduction 05/2012; 27(8):2297-303. · 4.47 Impact Factor
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ABSTRACT: Recurrent miscarriage is classically defined as three or more consecutive pregnancy losses. Established causes of recurrent
miscarriage are antiphospholipid antibodies, uterine anomalies and abnormal chromosomes in either partner, particularly translocations.
Embryonic aneuploidy is the most important cause of miscarriage before 10weeks’ gestation. It can be speculated that about
51% of patients with a history of three miscarriages experienced these because of abnormal embryonic karyotypes. It is not
necessary to give any medication for such cases caused by an abnormal embryonic karyotype. Psychological tender loving care
might be the most important requirement to continue conceiving till live birth results.
Reproductive Medicine and Biology 04/2012; 8(4):141-144.
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ABSTRACT: Career satisfaction level, degree of mental distress associated with certain work-related factors, and demographics were examined for the first time in obstetricians and gynecologists in Japan.
Associations between the score on Kessler 6 screening scale, or the job satisfaction level, and the scores on the job content questionnaire, Social Support Questionnaire (SSQ), working conditions and demographics were examined in 1301 members of the Japan Society of Obstetrics and Gynecology.
8.4% of respondents were speculated to suffer from depression or anxiety disorder. Multivariate linear regression analysis identified a heavier workload, less personal control, lower satisfaction on the SSQ, and longer working hours as being independent risk factors for mental distress.
Careful monitoring of the mental state is necessary for obstetricians and gynecologists with lower incomes, heavier workloads, lower degrees of personal control, and lower satisfaction scores on the SSQ.
Journal of Obstetrics and Gynaecology Research 03/2012; 38(3):550-8. · 0.94 Impact Factor
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ABSTRACT: Microangiopathic antiphospholipid-associated syndromes (MAPSs) are reported as encompassing several conditions mainly affecting the microvasculature of selected organs: the liver in HELLP syndrome (hemolysis, elevated liver enzymes, and low platelet); kidney, brain, and skin in TTP (thrombotic thrombocytopenic purpura). It is predominant in patients with catastrophic antiphospholipid syndrome (APS). A recent report suggests that APS is not only a thrombotic disease but also associated with microangiopathic features, and it can explain the greater prevalence of HELLP syndrome in these patients. We here report a case of MAPS during pregnancy associated with systemic lupus erythematosus (SLE) in early second trimester.
Case Reports in Medicine 01/2012; 2012:827543.
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ABSTRACT: This study reviews systematically the effectiveness of management strategies for carriers of a reciprocal translocation involving two chromosomes, ascertained on the basis of recurrent pregnancy loss. Subsequent pregnancy outcomes were tabulated based on whether management was medical or involved in vitro fertilization/preimplantation genetic diagnosis (IVF/PGD). A total of 129 cases from 13 articles met the criteria, of which 89% were managed medically. Before management, the overall live birthrate was 4% (19 of 484 pregnancies). Management was medical in 109 cases and IVF/PGD in 20 cases. Cumulative live birthrate was 74% (81 of 109 cases) in the medical management group and 35% (7 of 20) in the IVF/PGD group. Based on this systematic review, successful pregnancy outcomes are high following either medical management or IVF/PGD for carriers of a reciprocal translocation, ascertained on the basis of recurrent pregnancy loss. But it is difficult to compare outcomes directly for these two strategies because of the different end points reported. Understanding the differences is essential for effective counseling. Until a well-designed study comparing the two strategies is performed, or at least prospective cohort studies with strict entry criteria and definitions, the cumulative experience and success of both medical management and IVF/PGD must be used to counsel patients who are carriers of a reciprocal translocation, ascertained on the basis of recurrent pregnancy loss.
Seminars in Reproductive Medicine 11/2011; 29(6):470-81. · 3.80 Impact Factor
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ABSTRACT: Women with recurrent pregnancy loss have a 3.2 to 6.9% likelihood of having a major uterine anomaly and a 1.0 to 16.9% chance of having an arcuate uterus. Bicornuate and septate uterine have a negative impact on reproductive outcomes and are associated with subsequent euploid miscarriage. The impact of an arcuate uterus on pregnancy outcome remains unclear. There are no definitive criteria to distinguish among the arcuate, septate, and bicornuate uteri. The American Fertility Society classification of Müllerian anomalies is the most common standardized classification of uterine anomalies. According to estimates, 65 to 85% of patients with bicornuate or septate uteri have a successful pregnancy outcome after metroplasty. However, 59.5% of the patients with such anomalies have a successful subsequent pregnancy without surgery, with a cumulative live birthrate of 78.0%. There is no case-control study to compare live birthrates in women who had surgery compared with those who did not. Strict criteria to distinguish between the bicornuate and septate uterus should be established. Further study is needed to confirm the benefits of metroplasty.
Seminars in Reproductive Medicine 11/2011; 29(6):514-21. · 3.80 Impact Factor
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ABSTRACT: Hypophosphatasia is an inheritable disorder characterized by defective bone mineralization and a deficiency of tissue-nonspecific alkaline phosphatase (TNSALP) activity. Screening for mutations in the TNSALP gene allows genetic counseling and prenatal diagnosis of the disease in families with severe forms of hypophosphatasia. A 33-year-old, gravida 4, para 3 Japanese woman was referred to Nagoya City University Hospital for prenatal genetic counseling because of two previous occurrences of fetal bone anomalies. The molecular examination showed that the fetus was homozygous for the TNSALP gene mutation c.1559delT, each parent being heterozygous. Genetic counseling was offered and at the next pregnancy, chorionic villus sampling was performed, whereupon genetic analysis confirmed that the fetus did not carry the familial mutation c.1559delT. Postnatal molecular genetic analysis using the cord tissue can provide a diagnosis of lethal hypophosphatasia and prenatal genetic diagnosis of the TNSALP gene allows time for parental counseling and delivery planning.
Journal of Obstetrics and Gynaecology Research 05/2011; 37(10):1470-3. · 0.94 Impact Factor
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Journal of Obstetrics and Gynaecology Research 03/2011; 37(6):664-5. · 0.94 Impact Factor
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ABSTRACT: SYCP3 mutations have been shown to generate an aberrant synaptonemal complex in a dominant-negative manner and to contribute to abnormal chromosomal behavior that might lead to recurrent miscarriage. We examined whether SYCP3 mutation is associated with recurrent miscarriage caused by embryonic aneuploidy.
The SYCP3 657T>C mutation was examined using PCR and sequencing in 101 patients with a history of three or more unexplained recurrent miscarriages and 82 fertile controls with no history of miscarriage. The embryonic karyotype in the aborted conceptus was analyzed.
The 657T>C mutation of SYCP3 was identified in one patient with a history of six recurrent miscarriages with embryonic euploidy and one fertile woman in the control group. Patients with abnormal and normal chromosome were found to repeat miscarriage with abnormal and normal chromosome, respectively.
The 657T>C mutation of SYCP3 may not be associated with recurrent miscarriage caused by aneuploidy. We found no clinical significance of routine examination of the SYCP3 mutation because only one benign mutation was ascertained in 101 patients.
Human Reproduction 02/2011; 26(5):1259-66. · 4.47 Impact Factor
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American Journal of Medical Genetics Part A 12/2010; 152A(12):3189-92. · 2.39 Impact Factor
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ABSTRACT: The prevalence of antiphosphatidylethanolamine antibodies (aPEs) is higher in recurrent pregnancy loss patients than that in women with normal pregnancy. We conducted a cohort study to examine the predictive value of aPE for recurrent pregnancy loss and to determine its clinical significance. We examined plasma protein dependent (P+) and independent (P-) aPE IgG and IgM antibodies in 367 women with two or more unexplained consecutive pregnancy losses. We also examined conventional antiphospholipid antibodies (aPL) such as beta2-glycoprotein I-dependent anticardiolipin antibodies (beta2GPI-dependent aCL), lupus anticoagulant with reference to the dilute activated partial thromboplastin time (aPTT) and the diluted Russell's viper venom time (RVVT). Subsequent pregnancy outcome without medication was examined, and patients with and without aPE were compared. Totals of 37 (10.1%), 14 (3.8%), 23 (6.3%), 6 (1.6%), 9 (2.5%), 10 (2.7%) and 50 (13.6%) of the 367 patients were, respectively, positive for P+aPE IgG, P-aPE IgG, P+aPE IgM, P-aPE IgM, beta2GPI-dependent aCL, lupus anticoagulant by RVVT and LA by aPTT. The patients with aPE differed from patients with beta2GPI-dependent aCL or lupus anticoagulant by RVVT. No difference in live birth rate was apparent between positive and negative aPE patients with no medication. The areas under the curves for each ROC curve for the four aPEs were 0.535, 0.612, 0.546 and 0.533, respectively, so there was no significant variation in diagnostic capacity. We did not obtain any evidence that aPE elevation is an independent risk factor to predict further miscarriage in recurrent pregnancy loss patients.
Journal of Reproductive Immunology 06/2010; 85(2):186-92. · 2.97 Impact Factor
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International journal of gynaecology and obstetrics: the official organ of the International Federation of Gynaecology and Obstetrics 04/2010; 109(1):75-6. · 1.41 Impact Factor
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ABSTRACT: A 22-year-old pregnant woman noticed a rapid increase of abdominal growth, uterine tenderness and irregular contraction, for which she hospitalized at 25 weeks of gestation. An ultrasound examination demonstrated a single fetus with normal anatomy and massive hydramnios. Serial therapeutic amniocentesis was performed for relief of maternal symptoms and indomethacin compress was initiated. Both the maternal and amniotic fluid IgM were positive for cytomegalovirus (CMV). Maternal compress indomethacin was discontinued at 32 weeks. Cesarean section was performed due to fetal distress at 34 weeks of gestation. A female infant was delivered and the neonatal examination was within normal limits with urine culture positive for CMV. At 1 year of age the child was developing normally with normal hearing and no clinical sequelae of intrauterine CMV infection. We postulate that serial and large volume reduction of amniotic fluid by amniocentesis and compress indomethacin in our case interrupted the natural course and provided sufficient time for the fetus to recover from the acute phase of viral infection.
Congenital Anomalies 12/2009; 49(4):274-5.
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ABSTRACT: In Japan, marital age and women's age at the first pregnancy are continuing to increase year by year. However, information concerning subsequent live birth rate according to maternal age and number of previous recurrent miscarriages is limited.
We studied a total of 1250 unexplained patients suffering two or more consecutive miscarriages. We examined the live birth rate at the first pregnancy and the cumulative success rate for birth of at least one child after examination.
The live birth rate of women in their 40s was 58.1%, which was similar to that of women who were 35-39 years old (58.4%) at the first pregnancy, as found after examination. From logistic regression, women's age and the number of previous miscarriages independently decreased the live birth rate in subsequent pregnancies (p(s)) as well as cumulative pregnancies (p(c)), as follows: logit (p(s)) = 3.964 - 0.0652 x (age) - 0.408 x (previous number of miscarriages), logit(p(c)) = 6.806 - 0.1130 x (age) - 0.514 x (previous number of miscarriages).
The information concerning the live birth rate can be given to each patient before subsequent pregnancy.
American Journal Of Reproductive Immunology 11/2009; 62(5):314-9. · 2.17 Impact Factor
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ABSTRACT: We report four cases of persistent cloaca diagnosed at 32-33 weeks of gestation. In cases of persistent cloaca, serial prenatal ultrasonography shows transient fetal ascites, enlarged cystic structures arising from the fetal pelvis. Our four cases of persistent cloaca were diagnosed prenatally. Persistent cloaca should be considered in any female fetus presenting with hydronephrosis and a large cystic lesion arising from the pelvis as assessed by ultrasound and magnetic resonance imaging. Neither pulmonary hypoplasia nor severe oligohydramnios were found in any of our four cases, and they each had a good prognosis. Prenatal diagnosis allows time for parental counseling and delivery planning at a tertiary care center for neonatal intensive care and pediatric surgery.
Congenital Anomalies 09/2009; 49(3):116-7.
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ABSTRACT: To compare subsequent pregnancy outcomes after two or more miscarriages in patients with and without congenital uterine anomalies.
Case-control study.
Nagoya City University Hospital.
A total of 42 patients with a bicornuate or septate uterus and 1528 with normal uteri.
No surgery.
The cumulative success rate for birth, abnormal chromosome karyotype rate in aborted concepti, and the predictive values of the height of the defect/length of the remaining uterine cavity ratio (D/C ratio).
Of the total of 1676 patients, 54 (3.2%) had congenital uterine anomalies; 25 (59.5%) of the 42 patients with a bicornuate or septate uterus had a successful first pregnancy after examination, while this was the case for 1096 (71.7%) of the 1528 with normal uteri. There was no difference in the cumulative live-birth rate (78.0% and 85.5%) within the follow-up period. However, the rates for an abnormal chromosome karyotype in aborted concepti in cases with and without uterine anomalies were 15.4% (two of 13) and 57.5% (134 of 233), respectively, with the latter being significantly higher. The D/C ratio in the miscarriage group was also significantly greater than that for the live-birth group.
Congenital uterine anomalies have a negative impact on reproductive outcome in couples with recurrent miscarriage and are associated with further miscarriage with a normal embryonic karyotype. The D/C ratio was found to have a predictive value for further miscarriages in recurrent cases.
Fertility and sterility 03/2009; 93(6):1983-8. · 3.97 Impact Factor
