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1/64. A case of large placental chorioangioma with non-immunological hydrops fetalis.

    A 34-year-old Japanese woman (gravida 2, para 2) with polyhydramnios and non-immunological hydrops fetalis was referred to our department at 32 weeks of gestation. On admission, the blood pressure was 120/60 mmHg and there was no pitting edema of the lower extremities. An ultrasound examination disclosed a large placental tumor 5.8 cm x 4.4 cm x 4.8 cm. Fetal lung compression was suspected because the lung-thorax transverse area ratio was 0.13. The preload index of the inferior vena cava was 0.74, suggesting fetal cardiac failure. After fetal pleural effusion was aspirated, lung compression developed. cordocentesis was performed at 33 weeks of gestation, and the fetal karyotype was confirmed to be 46, XY from an umbilical blood cultivation. The patient underwent a cesarean section at 33 weeks of gestation due to severe uterine contraction after preterm PROM. The baby was a 3,840 g male with a distended abdomen. apgar score at 1 minute was 1. A chest X-ray demonstrated respiratory distress syndrome. The baby was discharged on the 69th day after birth and he is now 2 years and 9 months old and healthy.
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2/64. A patient with pseudohypoaldosteronism type 1 and respiratory distress syndrome.

    We present a patient born at 36 weeks' gestation with respiratory distress, who required 6 days of mechanical ventilation, without a demonstrable infectious cause. He also developed hyponatremia, hypernatriuria, elevated serum aldosterone levels, and probable pseudohypoaldosteronism type 1 (PHA-1). This appears to be the first reported human case of both respiratory distress and a renal salt wasting process with elevated serum aldosterone. In animal models, abnormalities of subunits of the epithelial sodium channel produce respiratory distress and PHA-1. This patient's clinical presentation could be due to the same processes.
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3/64. Complications of intrauterine intervention for treatment of fetal obstructive uropathy.

    The intrauterine surgical placement of vesicoamniotic shunts in the treatment of fetal obstructive uropathy associated with prune-belly syndrome to avoid such complications as renal damage and oligohydramnios remains controversial. We present a case of an infant born with prune-belly syndrome at 33 weeks and 5 days of estimated gestational age to a mother of two by vaginal delivery after a pregnancy complicated by fetal obstructive uropathy with attempted intrauterine intervention. After sonographic and laboratory diagnostic and prognostic evaluations, an intrauterine procedure was performed in which a vesicoamniotic shunt was placed under ultrasound guidance. Complications included dislodgment of the initial shunt, with a failed subsequent attempt at placement, oligohydramnios, preterm labor and delivery, and traumatic gastroschisis through the surgical abdominal wall defect. His hospital stay was further complicated by chronic renal insufficiency, prematurity, respiratory distress, bowel malrotation, an episode of gram-negative sepsis with enterobacter cloacae, signs of liver failure, an exploratory laparotomy for severe enterocolitis, and orchiopexy for bilateral undescended testes. At present, it is unclear whether vesicoamniotic shunt placement can provide any significant improvement in the morbidity or mortality for patients with prune-belly syndrome. A large, prospective, randomized trial is needed to determine its efficacy.
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keywords = gestation, pregnancy
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4/64. Complications of triple pregnancy following intracytoplasmic sperm injection: a case report.

    A case is presented of pregnancy and delivery of triplets following intracytoplasmic sperm injection (ICSI) therapy. Although the outcome was satisfactory, with the birth of normal children free from any malformation, most of the obstetric and particularly the neonatal complications that can be associated with this therapy are illustrated in this case. In addition, from point of view of medical costs, concerns are raised about the current policy of multiple embryo transfer which is directly responsible for the high rate of multiple gestations observed in the IVF/ICSI programme. The authors consequently recommend a policy of transferring not more than two embryos per treatment cycle.
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keywords = gestation, pregnancy
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5/64. A case report on the perinatal management of a 30-week preterm baby with congenital complete heart block.

    INTRODUCTION: Congenital complete heart block is an uncommon condition in the newborn, but is known to occur with maternal systemic lupus erythematosus. CLINICAL PICTURE: This paper presents one such baby with complete heart block who was born premature (after a gestation of 30 weeks) and weighing 759 g. TREATMENT: Continuous isoprnaline infusion was initially used to support the baby while her other neonatal problems were treated. A Medtronics VV1 pacemaker was subsequently inserted to maintain a heart rate that would be more physiologically acceptable for the patient. OUTCOME: This baby is currently thriving well, having been followed up for one year. CONCLUSIONS: The management issues, encompassing maternal and neonatal problems, and a review of current literature on this condition are discussed.
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6/64. One-sided high-frequency oscillatory ventilation in the management of an acquired neonatal lobar emphysema: a case report and review.

    We describe a premature infant (gestational age 28 weeks and birth weight 1280 g) with a left-sided acquired lobar emphysema (ALPE). Left lateral decubitus positioning, right-sided conventional ventilation (CV), tracheal high-frequency oscillatory ventilation (HFOV), and dexamethasone administration were subsequently used in the treatment without success. The emphysema was resolved and the patient was extubated after selective intubation and HFOV of the right unaffected lung. We also review the reported cases of ALPE in neonates that were treated by one-sided high-frequency ventilation (HFV).
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7/64. Neonatal diabetes mellitus: patient report and review of the literature.

    A female infant born at 33 weeks gestation to a gestationally diabetic mother developed apnea and respiratory distress at 6 hours of age. Laboratory investigation demonstrated persistent hyperglycemia, and the patient was treated with continuous intravenous and subsequent subcutaneous insulin therapy. Detailed laboratory investigation to reveal the etiology of hyperglycemia and further endocrine evaluation were not significant. The baby's insulin requirement has continued thereafter, and she is being followed up in an outpatient clinic still under insulin therapy at 18 months of age. Neonatal diabetes mellitus should be considered in the differential diagnosis of neonatal hyperglycemia, and it may develop in newborns born to diabetic mothers, as well as neonatal hypoglycemia. insulin treatment with close blood glucose monitoring is essential as long as hyperglycemia persists since neonatal diabetes mellitus may be either transient or permanent and it is not possible to differentiate these two outcomes before 18 months of age.
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8/64. Eradication of severe neonatal systemic candidiasis with amphotericin b lipid complex.

    OBJECTIVE: To report the successful use of amphotericin b lipid complex in treating severe systemic candidiasis in a very-low-birth-weight infant. CASE SUMMARY: A preterm female infant, born at 25 weeks' gestational age with a birth weight of 870 g, had received full supportive care in the neonatal intensive care unit (NICU), including mechanical ventilation, total parenteral nutrition, and placement of central venous catheters. At seven weeks of age, she developed severe disseminated candidiasis, which failed to respond to conventional amphotericin b and fluconazole therapy. Her progressive deterioration was reversed only after amphotericin b lipid complex (A-complex) was substituted for conventional amphotericin b. The improvement in her condition was impressive, and she made a full recovery without any adverse effect. DISCUSSION: With increased reliance on invasive technologies for life support, systemic candida infections have become increasingly common among premature infants in the NICU. Such infections are potentially fatal for the high-risk neonate. A literature review shows limited documentation of the use of lipid-based formulations of amphotericin b, especially A-complex, in preterm infants. However, the collective experience with these products appears to show that they are effective and cause fewer adverse effects than conventional amphotericin b. The infant reported here had shown progressive deterioration from disseminated candidiasis until conventional amphotericin b therapy was replaced with A-complex. Her recovery corresponded to the clearance of the candidemia. CONCLUSIONS: With favorable results and increasing experience with lipid-based formulations of amphotericin b, it is reasonable to consider these new formulations as therapy for candidemia in preterm infants who are at a high risk of nephrotoxicity or who have failed conventional therapy.
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9/64. trichosporon asahii: an unusual cause of invasive infection in neonates.

    trichosporon asahii causes white piedra, an infection of hair shafts and onychomycosis in immunocompetent patients, as well as various localized or disseminated invasive infections in immunodeficient hosts. We describe a 26-week gestation 890-g vaginally delivered female neonate who had severe respiratory distress syndrome and on the sixth day of life developed klebsiella pneumoniae sepsis. At the same time two blood cultures were positive for T. asahii. The neonate was also colonized with T. asahii in the pharynx and perineum. The infant was successfully treated with conventional amphotericin b.
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10/64. Inhaled nitric oxide for oligohydramnios-induced pulmonary hypoplasia: a report of two cases and review of the literature.

    We describe the clinical courses of two premature infants, a male born at 29(4/7) weeks' gestational age after an 8-week period of rupture of membranes (ROM) and severe oligohydramnios, and a female infant born at 31 weeks' gestational age after an 18-week period of ROM and severe oligohydramnios. Within hours after birth, despite intubation and aggressive ventilation, both infants developed fulminant hypoxic respiratory failure. Their clinical courses were consistent with pulmonary hypertension and both infants were transferred for trials of inhaled nitric oxide (iNO). Both infants had dramatic responses to iNO, suggesting that the pulmonary disease seen after prolonged oligohydramnios may have a component of nitric oxide-sensitive pulmonary hypertension. The goals of this article are to (1) review oligohydramnios-induced pulmonary hypoplasia, (2) discuss patients at highest mortality risk, and (3) describe the effects of iNO on pulmonary hypertension in infants with hypoxemia following prolonged ROM and severe oligohydramnios.
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