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THE JOURNAL OF THE JAPAN PEDIATRIC SOCIETY
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Vol.124, No.4, April 2020
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Review
Title
Serious Sequelae and Mortality with Subdural Hematoma Due to Low-Energy Trauma
Author
Ryo Sugitate Fumitake Mizoguchi and Atsushi Matsui
Department of Pediatrics, Maebashi Red Cross Hospital
Abstract
Recent studies in Europe and the US have re-evaluated and reported that low-energy forces can cause subdural hematoma (SDH), but this almost never leads to serious sequelae or death.
Japanese neurosurgeons conventionally used the Nakamura Type-I classification for SDH caused by low-energy forces, but the diagnosis was different between Japanese and Western reports. Recently, it has been criticized that the reports from foreign countries were accepted blindly, and false accusations have been made. Therefore, case reviews targeting medical and administrative reports in Japanese are needed to clarify the matter.
Japanese reports of medical literatures identified 144 cases of children below 2 years of age presenting SDH possibly caused by low-energy forces. Among them, mortality and permanent severe sequelae were noted in 5 and 10 cases, respectively, and a reliable third-party witness of the trauma was unavailable in all cases. In the reports by the Japanese administration, which were probably surveyed more extensively, there were no cases of SDH due to low-energy force accidents verified by a third-party witness.
Future prospective inventory survey studies are needed to verify the causal relationship between serious outcomes and SDH due to low-energy force accidents in pediatric patients.
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Original Article
Title
Retrospective Study of Intravenous Immunoglobulin during Plasma Exchange for Kawasaki Disease
Author
Hisakazu Majima1) Katsuaki Kasahara1) Sachiko Inukai2) and Yoshimitsu Gotoh1)
1)Department of Pediatric Nephrology, Japanese Red Cross Nagoya Daini Hospital
2)Department of Pediatrics, Japanese Red Cross Nagoya Daini Hospital
Abstract
Background
Plasma exchange (PE) is an effective treatment for refractory Kawasaki disease, but serum immunoglobulin G (IgG) level decreases after PE. We used to administer a single dose of intravenous IG (IVIG) after finishing the PE course, but since 2016, we have administered IVIG daily combined with PE. Here, we examined the effects.
Methods
Subjects comprised 34 patients who underwent PE for Kawasaki disease from January 2001 to December 2018. We retrospectively compared 24 patients in Group A who received IVIG as a single dose after the PE course with 10 in Group B who received IVIG daily during the PE course.
Results
Before starting PE, there was no statistically significant difference between groups. There was no significant between-group difference in the number of days of fever after PE initiation, the number of days until patients were discharged, and the number of patients with coronary artery aneurysm at 3 months after onset. However, the total IVIG dose was significantly higher in group B (p = 0.017).
Conclusion
IVIG daily administration during the PE course in patients with Kawasaki disease was more effective neither as an antipyretic nor in the prevention of coronary artery aneurysm than the administration of IVIG in a single dose after finishing the PE course. Given medical economics and potential adverse effects of IVIG administration, treatment by IVIG daily administration during the PE course in patients with Kawasaki disease is not recommended.
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Case Report
Title
Pregnancy and Delivery with Fontan Circulation Complicated by Critical Obstetric Hemorrhage and Fetomaternal Blood Transfusion Syndrome
Author
Fumitake Miura1) Madoka Fujita1) Kentaro Yuzawa1) Shun Koyamaishi1) Jun Shimada1) Yosuke Kitagawa1) Katsuki Otani1) Manabu Kinjo2) Toru Takahashi3) and Etsuro Ito1)
1)Department of Pediatrics, Hirosaki University School of Medicine and Graduate School of Medicine
2)Department of Pediatrics, Hachinohe City Hospital
3)Hirosaki University School of Health Sciences and Graduate School of Health Sciences
Abstract
There are few reports on pregnancy and delivery with Fontan circulation. Furthermore, there are no established guidelines on anticoagulation therapy in the perinatal period of pregnancy and during delivery with Fontan circulation. The patient was a primipara with tricuspid valve atresia (IIc), and Fontan circulation was performed at 8 years of age. She developed multiple cerebral infarctions during the postoperative course of Fontan operation. She had a natural pregnancy at 27 years of age. At 27 weeks of gestation, we diagnosed fetal anemia upon examination; consequently, she underwent an emergency cesarean section and delivered a boy weighing 1050 g. He developed fetomaternal blood transfusion syndrome. The mother was administered anticoagulation treatment approximately 7 hours after the caesarean section. However, she abruptly developed a hematoma in the abdominal wall, causing critical obstetric hemorrhage. An emergency surgical evacuation of the hematoma was performed. Placental pathology revealed chronic circulatory failure. Thus, failure of the placental circulation must be considered in such cases. We should also consider thrombotic and hemorrhagic complications when managing pregnancy and delivery with Fontan circulation.
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Case Report
Title
Psychological Stress-Induced Acute Exacerbation of Non-Neurogenic Bladder with Down's Syndrome
Author
Mari Okada1) Hisae Nakatani1) Keiko Onda1) Masako Imai1) Natsuko Suzuki1) Masayuki Nagasawa1) Yoshinori Tanaka2) Akihiro Oshiba1) and Masuhiro Shimoda3)
1)Department of Pediatrics, Musashino Red Cross Hospital
2)Department of Urology, Musashino Red Cross Hospital
3)Department of Pediatrics, Nerima Hikarigaoka Hospital
Abstract
Herein, we report the case of a 13-year-old boy with Down's syndrome who had acute kidney injury caused by urinary retention following traumatic stress. The patient suffered from loss of appetite and dysuria. His serum creatinine was elevated, and the ultrasound revealed bilateral hydronephrosis and bladder wall thickening. We diagnosed the patient with postrenal acute kidney injury and proceeded to insert a urinary catheter. After placement, renal function improved gradually. We performed a voiding cystourethrogram and identified a bladder diverticulum and bladder wall irregularity. According to imaging analysis, it was possible that the patient experienced dysuria from a very young age. His parents noticed he had abdominal straining during voiding since he was 6 years old. However, they never consulted a physician. There are some reports on lower urinary tract disorders associated with Down's syndrome. However, guidelines on Down's syndrome published by the American Academy of Pediatrics do not offer instructions on follow-up with lower urinary tract disorders. We propose that physicians who treat Down's syndrome patients should be made aware of this requirement.
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Case Report
Title
A 12-Month-Old Infant Diagnosed with Familial Hypercholesterolemia Secondary to the Investigation of His Father's Angina Pectoris
Author
Hikaru Nagaseki1) Masaharu Akiyama1) Mika Hori2) Mariko Shiba2) Hiroyuki Ida1) and Ichiro Miyata1)
1)Department of Pediatrics, The Jikei University School of Medicine
2)Department of Molecular Innovation in Lipidology, National Cerebral and Cardiovascular Center Research Institute
Abstract
Familial hypercholesterolemia (FH) is a genetic disorder caused by mutations in the low-density lipoprotein receptor (LDL-R) and other related genes. Although the frequency of homozygous FH is 1 in 160,000 to 1 in a million individuals, heterozygous FH is found in 1 in 200 to 1 in 500 individuals. These patients are, therefore, regularly encountered in daily clinical practice. Patients with homozygous FH display clinical signs such as xanthomas and symptoms of premature coronary atherosclerosis. In contrast, patients with heterozygous FH are asymptomatic, making early diagnosis challenging. This case reports on the diagnosis of a 12-month-old boy with FH, after the industrial doctor referred his father to our hospital for investigation of his angina pectoris and dyslipidemia, with paternal grandfather's medical history. Although our patient was asymptomatic, his blood analysis revealed high levels of serum total cholesterol and LDL-cholesterol. Genetic analysis revealed that both he and his father have a novel heterozygous duplication of exon 8 to 10 in the LDL-R gene. Aside from the heterozygous mutation, the large duplication present over several exons may cause an abnormal structure of the LDL-R protein, resulting in their FH. Nutritional control alone was insufficient to lower his serum LDL-cholesterol levels, and he is currently receiving cholestyramine treatment.
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