Successful intra-amniotic levothyroxine treatment for fetal goitrous hypothyroidism in a triplet pregnancy.

We report an extremely challenging case of fetal goitrous hypothyroidism involving all three fetuses of a triplet pregnancy in which successful fetal treatment led to a favorable pregnancy outcome. The patient had a trichorionic, triamniotic triplet pregnancy and was referred to us at 24 weeks gestation after goiters affecting all three fetuses and polyhydramnios involving two fetuses were noted. Immediately before the conception, she underwent hysterosalpingography with an oil-soluble iodinated contrast medium. After the diagnosis of fetal hypothyroidism was made, intra-amniotic injection of levothyroxine was performed for two fetuses with polyhydramnios 3 times between 28 and 31 weeks gestation. The goiters shrunk and the polyhydramnios improved in response to the in utero treatment. No complications occurred. Cesarean section was performed at 33 weeks gestation. None of the three neonates developed respiratory insufficiency. Our experience suggested that successful intrauterine treatment is possible for fetal goitrous hypothyroidism, even in a triplet pregnancy. The indication, treatment timing, and diagnostic and assessment strategies should be carefully discussed to minimize puncture-related complications.

A Rare Case of Fetal Cardiac Hypertrophy Developing into Acute Circulatory Insufficiency and Fetal Compromise in Type 1 Diabetic Pregnancy.

Fetal cardiac hypertrophy (CH) in pregnant women with diabetes is believed to be a benign condition. We encountered a rare case of fetal CH in a pregnant woman with type 1 diabetes, which developed into severe fetal circulatory insufficiency and acidemia. Fetal echocardiography at 37-week gestation showed cardiomegaly with a ventricular hypertrophy. Cardiac function was impaired, and pulsed Doppler findings indicated circulatory failure. The patient was diagnosed with fetal compromise due to fetal CH, and a large for gestational age boy was delivered by an urgent cesarean section. Despite myocardial hyperplasia and left ventricular outflow tract stenosis, the neonate was hemodynamically stabilized by fluid resuscitation alone. Although the neonatal course was favorable, we speculated that the neonate was on the verge of death because he was already acidemic at birth. Therefore, comprehensive fetal echocardiography should be performed in pregnant women with diabetes, and clinicians should not miss the optimal timing of delivery.

Intrathoracic shunt displacement with massive pleural effusion after successful shunt placement in a hydropic fetus with multilocular macrocystic congenital pulmonary airway malformation.

We successfully performed shunting for a fetus with a multilocular macrocystic lung mass with hydrops at 22 weeks' gestation. Complete resolution of hydrops was achieved; however, at 35 weeks' gestation, the fetus developed acute massive pleural effusion. Fetal ultrasound examination revealed that one end of the shunting tube had migrated downward in the thoracic cavity, which led to fluid draining from the lung cyst. The baby was delivered at term and was discharged following neonatal intensive care management. Intrathoracic displacement of the shunt can occur, followed by massive pleural effusion due to drainage of cystic fluid.

The impact of general anesthesia on mother-infant bonding for puerperants who undergo emergency cesarean deliveries.

Background Mother-infant bonding is an emerging perinatal issue. While emergency cesarean deliveries are associated with a risk of bonding disorders, the mode of anesthesia used for emergency cesarean deliveries has never been studied in this context. We aimed to investigate the impact of administering general anesthesia and neuraxial anesthesia to women undergoing cesarean deliveries on mother-infant bonding. Methods This was a retrospective, propensity score-matched multivariable analysis of 457 patients who underwent emergency cesarean deliveries between February 2016 and January 2019 at a single teaching hospital in Japan. The Mother-Infant Bonding Scale (MIBS) scores at hospital discharge and the 1-month postpartum outpatient visit were evaluated in the general anesthesia and the neuraxial anesthesia groups. A high score on the MIBS indicates impaired mother-infant bonding. Results The primary outcome was the MIBS score at hospital discharge in propensity score-matched women. After propensity score matching, the median [interquartile range (IQR)] MIBS scores were significantly higher in the general anesthesia group than those in the neuraxial anesthesia group at hospital discharge [2 (1-4) vs. 2 (0-2); P = 0.015] and at the 1-month postpartum outpatient visit [1 (1-3) vs. 1 (0-2); P = 0.046]. In linear regression analysis of matched populations, general anesthesia showed a significant and positive association with the MIBS scores at hospital discharge [beta coefficient 0.867 (95% confidence interval [CI] 0.147-1.59); P = 0.019] but not at the 1-month postpartum outpatient visit [0.455 (-0.134 to 1.044); P = 0.129]. Conclusion General anesthesia for emergency cesarean delivery is an independent risk factor associated with impaired mother-infant bonding.

Massive atonic bleeding during cesarean delivery in a patient with chronic idiopathic intestinal pseudo-obstruction: A case report and literature review.

A 35-year-old primigravid woman with chronic idiopathic intestinal pseudo-obstruction presented to our institution. Except for an enlarged fetal bladder, her pregnancy was almost uneventful until she developed pre-eclampsia requiring emergent cesarean section at 34 weeks gestation. After delivery, intractable uterine atony developed with blood loss reaching 3500 mL within 15 min. Following a B-Lynch suture, the bleeding attenuated but uterine atony persisted; lochia persisted for 3 months post-partum. The infant was diagnosed with megacystis microcolon intestinal hypoperistalsis syndrome after birth. The mother's clinical course and previous reports suggested that atonic bleeding was associated with the pathology of chronic idiopathic intestinal pseudo-obstruction; the infant's disease was considered to be maternal-related disease. Clinicians should be vigilant in pregnant patients with chronic idiopathic intestinal pseudo-obstruction especially with these complications.

Fibrosis in Preeclamptic Placentas Is Associated with Stromal Fibroblasts Activated by the Transforming Growth Factor-ß1 Signaling Pathway.

Although fibrosis is one of the most prominent pathologic features of preeclamptic (PE) placentas, its mechanism remains largely unknown. Consistent with previous reports, we observed overexpression of collagen; actin, α2, smooth muscle, aorta; connective tissue growth factor; and fibronectin in PE placentas compared with control ones. To investigate the mechanism of fibrosis in PE placentas, placental fibroblasts were isolated from PE placentas or normal pregnancies at delivery. The expression of fibrosis-related factors in fibroblasts was evaluated by real-time RT-PCR, Western blotting, enzyme-linked immunosorbent assay, and gene microarrays. An in vitro collagen gel contraction assay was also performed. Fibroblasts isolated from PE placentas showed higher expression levels of fibrosis-related factors compared with those from control ones. Global gene expression profiling of PE fibroblasts was contrasted with that of control ones and indicated an intimate association with transforming growth factor-ß1 (TGFB1) signaling. Furthermore, the PE fibroblasts expressed abundant phosphorylated SMAD family member 2 and showed higher expression levels of target genes of TGFB1 signaling compared with the control ones. The PE fibroblasts also had a greater ability to contract compared with the control ones. Contractility also depended on TGFB1 signaling. Our results suggest that TGFB1 signaling is activated in the fibroblasts in PE placentas and that these active fibroblasts contribute to fibrosis.

Prolongation of Second Twin's Delivery Until Term: A Rare Case of Delayed-Interval Delivery.

Although some cases describing delayed birth of the second twin have been published recently, delay of delivery beyond 36 weeks seems scarce. We report a case of delayed-interval delivery wherein prolongation of the second twin's delivery until term with a subsequent favorable infantile outcome. In this case, the stillbirth of the first twin occurred at 25 weeks' gestation. Prophylactic tocolysis was performed with ritodrine and magnesium sulfate, and a McDonald cerclage was performed 2 days after delivery of the first twin. Ampicillin and gentamicin were also administered for the purpose of prevention of intrauterine infection. No clinical sign of chorioamnionitis was found thereafter, and full term uneventful delivery was achieved. With this experience, we believe that delayed-interval delivery can be effective in prolonging gestation and should be an option if the first twin developed an extreme preterm delivery.

A Rare Case of Fetal Meconium Peritonitis Developing Coagulopathy In utero.

Hydrops fetalis in association with meconium peritonitis is a rare condition, and the mechanism underlying hydropic changes has not been fully recognized. We present a case of fetal meconium peritonitis with hydrops and coagulopathy. Clinically, the cause of fetal disseminated intravascular coagulation is considered to be a consequence of a systematic inflammatory response based on progressive, but mild fetal anemia without other apparent triggers, thrombocytopenia, elevated white blood cell count and serum C reactive-protein, hypoalbuminemia, and increased vascular permeability. The infant was born at 32 weeks of gestation and survived after postnatal multidisciplinary treatment. Our experience suggests that recognition of this rare condition will enable early diagnosis and better clinical management for fetuses with meconium peritonitis.

Expectant management of pregnancies complicated by fetal growth restriction without any evidence of placental dysfunction at term: Comparison with routine labor induction.

AIM: To assess the feasibility and practicality of expectant management for pregnancies with fetal growth restriction (FGR) at term without evidence of placental dysfunction. METHODS: We reviewed the records of pregnancies with an estimated fetal weight ≤ 1.5 SD below the mean at 37 weeks of gestation. We excluded elective cesarean deliveries and pregnancies that, at 37 weeks, were complicated by oligohydramnios, decreased fetal cerebroplacental ratio, or pregnancy-related hypertensive disorders. Prior to May 2013, we performed routine labor induction for FGR at term; after that time, we used routine expectant management. The rate of delivery by cesarean or instrumental assist and the rate of neonatal morbidity were compared between the groups. RESULTS: The gestational age at delivery and the neonatal birthweight were higher in the expectant management policy group (39+4 vs 38+1 weeks; 2405 vs 2205 g). The cesarean rate (7/77 vs 7/73) and the instrumental delivery rate (5/77 vs 6/73) did not differ. Neonatal hypoglycemia and hyperbilirubinemia were significantly less frequent (10/77 vs 21/73; 7/77 vs 20/73) in the expectant management policy group. Seven patients in the expectant management policy group underwent emergency cesarean delivery; five of these (71%) had required labor induction because of progression to oligohydramnios. CONCLUSIONS: Expectant management policy for FGR at term can reduce neonatal morbidity without increasing maternal risk or the cesarean rate. Caution should be used, however, during labor if oligohydramnios develops during expectant management.

Oncologic and obstetric outcomes and complications during pregnancy after fertility-sparing abdominal trachelectomy for cervical cancer: a retrospective review.

BACKGROUND: Trachelectomy was developed as a fertility-sparing surgery for early-stage cervical cancer in patients of childbearing age. The purpose of this study is to evaluate oncologic and obstetric outcomes and complications after abdominal trachelectomy. METHODS: We began to perform abdominal trachelectomy in 2005. Our institutional review board approved this clinical study, and fully informed consent was obtained from each patient. The medical records of patients who underwent trachelectomy were retrospectively reviewed. RESULTS: We performed 151 abdominal trachelectomies (89 radical trachelectomies, 48 modified radical trachelectomies, and 14 simple trachelectomies). The median age of the patients was 33 years, and the median postoperative follow-up period was 61 months. Although one patient experienced recurrence at the preserved cervix, none died after treatment. A total of 61 patients attempted to conceive after trachelectomy, and 21 pregnancies were achieved in 15 women. Hence, the pregnancy rate among patients who attempted to conceive was 25%. Fifteen babies were delivered by cesarean section between gestational weeks 23 and 37. Six babies were delivered at term. Six cases of preterm premature rupture of the membranes occurred. Varices appeared around the uterovaginal anastomotic site in five patients. CONCLUSIONS: Our data indicate that the oncologic outcome was excellent but infertility treatment was necessary to achieve the majority of conceptions. Additionally, preterm premature rupture of the membranes and premature delivery were frequently observed. An improved pregnancy rate and prevention of complications during pregnancy are issues that should be addressed in future studies.

Associated anomalies in congenital diaphragmatic hernia: perinatal characteristics and impact on postnatal survival.

OBJECTIVE: We aimed to investigate the clinical characteristics and postnatal outcomes of fetuses with congenital diaphragmatic hernia (CDH) and additional anomalies. MATERIALS AND METHODS: We reviewed the charts of fetuses with CDH managed between 2005 and 2013. Patients were divided into complex and isolated groups based on the presence of additional anomalies. We analyzed the respective polyhydramnios, liver herniation, stomach position, lung to thorax transverse area ratio (LTR), and prognoses of the two groups. The survival rates of both groups were assessed based on the LTR as well as on stomach and liver positions. RESULTS: CDH was diagnosed in 65 fetuses, and additional anomalies were found in 23. The incidences of liver herniation, polyhydramnios, and death were significantly higher, and LTR was significantly lower, in the complex group. The mortality rate of fetuses with a LTR <0.08 was lower than that of fetuses with a LTR of ≥0.08 in the complex group. Further, the survival rate of fetuses with intrathoracic liver was lower than those without liver herniation. CONCLUSIONS: The prognosis of complex CDH is poor. This may result from both the associated anomalies and the severity of CDH itself. Even in complex CDHs, intrathoracic liver and LTR values are useful in estimating postnatal outcome.

Spontaneous post-partum rupture of an ovarian artery aneurysm: a case report of successful embolization and a review of the published work.

It is rare for an ovarian artery aneurysm to rupture during the peripartum period, but the outcome can be catastrophic. Between 1980 and 2013, only nine cases have been reported in the English-language medical published work. We describe a patient with a left ovarian artery aneurysm that ruptured 4 days after an uneventful vaginal delivery. The rupture was immediately identified, using emergent computed tomography and angiography. The hemorrhage was managed using arterial embolization and blood transfusion, allowing for a successful outcome. The possibility of ruptured ovarian artery aneurysm should be considered in the differential diagnosis for women with excessive or localized abdominal pain after childbirth.

External iliac artery thrombosis after common iliac artery balloon occlusion during cesarean hysterectomy for placenta accreta in cervico-isthmic pregnancy.

Although the role of interventional radiology in the field of obstetrical hemorrhage has been widely reported upon recently, the rate of procedure-related complications has not been fully determined. We present the case of a patient who developed an external iliac artery thrombosis, a rarely reported complication associated with prophylactic common iliac artery balloon occlusion (CIABO). After CIABO, we found that the dorsalis pedis artery of the right foot was weak and the foot was cold, despite the fact that the patient had no complaints. Computed tomography demonstrated a linear thrombus in the right external iliac artery. We managed the patient conservatively using a heparin drip without the need for thromboembolectomy. Our experience suggests that it is important to consider the risk of thrombosis formation after CIABO. Physical examination post-procedure is key to identifying this complication early.

Monochorionic monozygotic twin pregnancy complicated with twin-twin transfusion syndrome presenting with an obvious lambda sign in the first trimester.

In this report, we present a case of twin-twin transfusion syndrome in the presence of the thick dividing wall of each sac in dichorionic twins. The dichorionic diagnosis was based on the presence of the lambda sign at the first-trimester ultrasound evaluation. In addition to fetoscopic and pathological investigation, DNA typing confirmed that the twin set was monochorionic and monozygotic. This case illustrates that although extremely uncommon, the presence of lambda sign does not exclude monochorionic pregnancy.

In utero resolution of microcystic congenital cystic adenomatoid malformation after prenatal betamethasone therapy: A report of three cases and a literature review.

Fetal congenital cystic adenomatoid malformation (CCAM) can progress to nonimmune hydrops, and the mortality rate of CCAM with hydrops is reported to be nearly 100%. We describe three microcystic CCAM cases in which the fetal condition improved after maternal betamethasone therapy. The median gestational age at steroid administration was 23 5/7 weeks' gestation. The CCAM decreased in size in all cases. Our series showed a 100% hydrops resolution rate (2/2) and a 100% survival rate (3/3). Our experience suggests the efficacy of betamethasone treatment on fetuses with microcystic CCAM who have fluid collection or are at risk of developing hydrops.

Magnetic resonance fetal right lung volumetry and its efficacy in predicting postnatal short-term outcomes of congenital left-sided diaphragmatic hernia.

AIM: We aimed to investigate whether the ratio of magnetic resonance imaging (MRI)-measured right lung volume (RLV) to ultrasonography-estimated bodyweight (RLV/BW) and observed-to-expected (o/e) RLV are of diagnostic value in predicting postnatal outcomes of left congenital diaphragmatic hernia (CDH). MATERIAL AND METHODS: We included 32 CDH patients and 34 control subjects. Manually outlined fetal right lung areas on MRI were multiplied by the slice thickness and added to determine the entire volume. The association between RLV and RLV/BW with gestational age in the controls was examined using regression analysis. RLV/BW and o/e RLV were compared between surviving and non-surviving neonates with CDH. RESULTS: The expected fetal RLV was derived using the formula RLV (mm(3)) = 1.717 × (gestational weeks)(2.82). In the controls, RLV/BW was nearly constant during the third trimester. The 27 survivors with CDH had a median RLV/BW of 10.7 and a median o/e RLV of 60.0, whereas the five non-surviving neonates had a median RLV/BW of 4.3 and a median o/e RLV of 22.6; the differences were statistically significant. CONCLUSION: Assessment of fetal lungs by MRI volumetry is reliable for clinical use. RLV/BW and o/e RLV are potential predictors of postnatal outcomes of left CDH.

Successful ex utero intrapartum treatment procedure for prenatally diagnosed severe micrognathia: a case report.

We present a case of fetal severe micrognathia in which successful airway stabilization was achieved by an ex utero intrapartum treatment procedure. In this case, it was anticipated that the infant would have a vulnerable airway at birth based on in utero sonographic findings, including an extremely hypoplastic jaw, worsening polyhydramnios and absence of stomach visualization. Early sonographic recognition was helpful in preparing the parents and physicians for the possibility of airway emergencies during the perinatal period. When a severely hypoplastic mandible accompanied by polyhydramnios and absent stomach visualization is noted on ultrasound, clinicians should consider the indication for ex utero intrapartum treatment. A multidisciplinary team with technically skilled medical providers should be coordinated to perform the procedure.

Perinatal characteristics of fetuses with borderline ventriculomegaly detected by routine ultrasonographic screening of low-risk populations.

AIM: Fetal borderline ventriculomegaly represents a frequent dilemma in perinatal management. The present study aimed to evaluate the clinical significance of fetal borderline ventriculomegaly in a low-risk Japanese population and to identify the risk factors for associated anomalies. METHODS: Data of cases of fetal borderline ventriculomegaly detected at 26-28 weeks of gestation by routine ultrasonographic screening of low-risk singleton pregnancies between 2006 and 2012 were retrospectively collected. Ventricular width, in utero progression, associated anomalies, chromosomal abnormalities, and perinatal and postnatal outcomes were assessed. The ventricular width, in utero progression and other perinatal characteristics were compared between the isolated and non-isolated groups. RESULTS: Among the total 6020 singleton low-risk pregnancies, we noted that 42 had borderline ventriculomegaly. Six (14%) of these cases had other defects by subsequent detailed examination. Ventriculomegaly resolved or regressed in 35 (83%) and progressed in four (10%) cases, of which three were associated with other anomalies. The median ventricular width was 12.8 mm (range, 10.0-14.7) in the six non-isolated cases and 10.5 mm (range, 10.0-13.3) in the 36 isolated cases; the differences were statistically significant. A ventricular width of 12 mm or more and in utero progression were more frequently observed in non-isolated cases than in isolated cases. CONCLUSION: Fetal borderline ventriculomegaly frequently resolves in utero. A ventricular diameter of more than 12 mm and in utero progression are risk factors for additional anomalies. After the initial diagnosis of borderline ventriculomegaly, the pregnancy should be carefully followed up to determine whether the ventricle size is resolved, remains stable or increases.

Significance of maternal screening for toxoplasmosis, rubella, cytomegalovirus and herpes simplex virus infection in cases of fetal growth restriction.

AIM: The objective of this study was to evaluate the significance of maternal toxoplasmosis, rubella, cytomegalovirus (CMV) and herpes simplex virus (TORCH) screening in cases of fetal growth restriction (FGR). MATERIAL AND METHODS: The medical records of women carrying fetuses with FGR who underwent TORCH screening over a 10-year period were retrospectively reviewed for maternal and congenital TORCH infection. Women carrying fetuses with FGR routinely underwent serologic TORCH tests and systematic ultrasound evaluation for congenital abnormalities. If a congenital CMV infection was suspected, amniotic fluid, placenta or neonatal urine was used for CMV DNA detection by polymerase chain reaction. RESULTS: In 319 patients, no cases of maternal or congenital infection with toxoplasma, rubella, or herpes simplex virus were found. Conversely, six cases (1.8%) were diagnosed with congenital CMV infection, two of which had no structural abnormalities other than FGR. CONCLUSIONS: A complete maternal TORCH screening for cases of FGR appears to be unnecessary. Although a maternal CMV test can be considered, the incidence of congenital CMV infection was found to be low in FGR cases.

Fetoscopic diagnosis of congenital megalourethra at early second trimester.

Congenital megalourethra is a rare urogenital malformation that is usually associated with lower urinary tract obstruction. In association with hypoplasia or deficiency of the corpus spongiosa and/or corpus cavernosum, a cystic dilatation of penile urethra is generally observed. We present a case of congenital megalourethra diagnosed by fetoscopy and ultrasonography at 17 weeks of gestation. Fetoscopic investigation demonstrated a balloon dilatation of the penis with a complete obstruction of the urethral meatus, which could distinguish a fusiform type congenital megalourethra from other disorders. Autopsy subsequent to the termination of pregnancy revealed a complete deficit of both corpus spongiosa and corpus cavernosum.