Brain injury following mild hypoxic-ischemic encephalopathy in neonates-Ten-year experience in a tertiary perinatal center.

OBJECTIVE: We aimed to evaluate abnormal magnetic resonance imaging (MRI) findings in neonates with mild hypoxic-ischemic encephalopathy (HIE). STUDY DESIGN: This was a single-center, retrospective study of mild HIE conducted from 2011 to 2020. Almost all neonates with mild HIE received management targeted to a temperature of 36 ± 0.5°C for 72 h and underwent MRI (135 of 145). We evaluated medical records and assessed amplitude-integrated electroencephalography (aEEG). RESULT: All participants survived until discharge. Abnormal MRI findings were present in 23/135 (17%) neonates. The normal versus abnormal MRI-finding groups had similar characteristics, except discontinuous aEEG patterns (p = 0.002) and inotropic drugs (p = 0.035). Regression analyses showed discontinuous aEEG patterns (odds ratio = 19.3, 95% confidence interval=1.88-197, p = 0.013) being associated with higher odds of abnormal MRI findings. CONCLUSION: Our study suggests that a discontinuous aEEG pattern is associated with abnormal MRI findings and can be used to discuss the definition of mild HIE.

Fetal-onset malignant rhabdoid tumor: a case report.

BACKGROUND: A fetal-onset cervical mass may cause postnatal airway obstruction, and ex utero intrapartum treatment (EXIT) to secure the airway while maintaining fetal-placental circulation may be life-saving. Malignant rhabdoid tumors (MRT) are highly aggressive tumors, and when they develop in utero, the prognosis is even worse, with almost no reports of survival beyond the neonatal period. Herein, we report a case of a primary cervical MRT and describe our treatment using EXIT for securing the airway, wherein the infant's life was saved. CASE PRESENTATION: A 40-year-old Japanese woman with no relevant medical or surgical history was diagnosed with a fetal left cervical mass and polyhydramnios during the third trimester. Fetal magnetic resonance imaging indicated the possibility of postnatal airway obstruction, and delivery using EXIT was planned. The infant was delivered by a planned cesarean section at 39 weeks and 5 days gestation, and tracheostomy was performed using EXIT. Postnatal contrast-enhanced computed tomography revealed suspected metastatic lesions in the subcutaneous tissue, lungs, and thymus, in addition to the mass in the left cervical region. MRT was diagnosed by biopsy of a subcutaneous mass in the left thigh, and chemotherapy with vincristine, doxorubicin, cyclophosphamide, ifosfamide, and etoposide was initiated. The tumors regressed, and the infant was successfully weaned from artificial ventilation. After discharge from the hospital, she had a recurrent cervical mass and intracranial metastasis, and radiotherapy was initiated. CONCLUSIONS: In our case, fetal diagnosis enabled advance planning of delivery using EXIT, thus saving the infant's life. The use of chemotherapy for MRT, which has a poor prognosis, allowed tumor regression and enabled the infant to survive beyond the neonatal period.

The Relationship between Serum Total Bilirubin and Severity of Hypoxic Injury in Neonatal Hypoxic-Ischemic Encephalopathy.

INTRODUCTION: Bilirubin has been found to be a natural antioxidant protecting the body from oxidative stress. This study aims to investigate the severity of neonatal hypoxic injury on bilirubin levels to clarify the physiological role of bilirubin. STUDY DESIGN: Using lactate as a biomarker for the severity of hypoxic injury, we retrospectively analyzed the association of admission lactate levels on serum total bilirubin (TB) levels following birth in neonates with hypoxic-ischemic encephalopathy (HIE). We created a univariate linear regression model to predict TB using the admission lactate level as a predictor. We also performed a multivariate linear regression analysis to predict TB with admission lactate levels that included phenobarbital cumulative dosage, admission hematocrit, and subgaleal hemorrhage as predictor variables. RESULTS: A total of 86 patients were studied. Admission lactate levels had a significant negative effect on TB from admission to 3 days of life. The standardized regression coefficient for admission lactate on TB was -0.37 (admission), -0.42 (day 1), -0.32 (day 2), and -0.28 (day 3). A similar negative effect of admission lactate on TB levels was also observed in the multivariate linear regression model even after controlling for the other variables. CONCLUSION: These results are consistent with the hypothesis that bilirubin functions as an antioxidant in vivo, and is consumed by scavenging free radicals in proportion to the severity of hypoxic injury. KEY POINTS: · Lactate levels have a negative effect on serum TB levels in patients with neonatal HIE.. · Bilirubin might work as a natural antioxidant to protect the body from oxidative stress.. · Serum TB might be a marker for the severity of hypoxic injury..

Mitochondrial Disease as a Cause of Neonatal Hemophagocytic Lymphohistiocytosis.

Diagnosis of mitochondrial respiratory chain disorder (MRCD) is often difficult. Its pathogenesis is still unclear. We diagnosed MRCD by measuring the activity of the mitochondrial respiratory chain enzyme, and the patient also had hemophagocytic lymphohistiocytosis (HLH). A preterm female infant was born at 34 weeks of gestation. On day 6, HLH was revealed by bone marrow aspiration. She died on day 10 due to uncontrollable HLH. An autopsy was performed, and we measured the activity of the mitochondrial respiratory chain enzyme in the liver, muscle, and heart. The activity of complex I was decreased in all tissues. As we could not prove another origin of the HLH, she was diagnosed as having HLH caused by MRCD. It is useful to measure the activity of the mitochondrial respiratory chain enzyme for diagnosing MRCD. MRCD, which has a severe clinical course, may be related to HLH.

[Safety and efficacy of surgical closure of the larynx for recurrent aspiration pneumonia in persons with severe motor and intellectual disabilities: a comparative study with tracheoesophageal diversion].

OBJECTIVE: We retrospectively investigated the efficacy and complications of surgical closure of the larynx (SCL) for recurrent aspiration pneumonia in comparison with tracheoesophageal diversion. METHODS: The subjects were persons with severe motor and intellectual disabilities (SMID) who had undergone surgery for recurrent aspiration pneumonia between 1994 and 2011: A 8 SCL patients group and a 16 tracheoesophageal diversion patients group. We investigated two groups the lower respiratory infection incidence, length of hospital stay for the surgery, postoperative complications, and rate of cannula withdrawal, by reviewing medical records. RESULTS: Both the SCL and the tracheoesophageal diversion group showed a reduction in the incidence of infection after surgery, indicating that the efficacy of SCL was equivalent to that of tracheoesophageal diversion in preventing aspiration pneumonea. The SCL group showed a reduction in the length of hospital stay and an increased rate of cannula withdrawal as compared with the tracheoesophageal diversion group. CONCLUSION: The efficacy of SCL was equivalent to that of tracheoesophageal diversion in preventing aspiration for SMID. We consider SLC to have potential for reducing the burden on patients.