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THE JOURNAL OF THE JAPAN PEDIATRIC SOCIETY
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Vol.126, No.1, January 2022
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Review
Title
A Scoping Review of Pediatric Economic Evaluation in Japan
Author
Kimiko Honda1) and Takashi Fukuda1)2)
1)Department of Public Health Science, School of Public Health, The University of Tokyo
2)Center for Outcomes Research and Economic Evaluation for Health, National Institute of Public Health
Abstract
Economic evaluation is widely used to ensure the proper allocation of medical resources in many countries, as national healthcare costs continue to increase. In Japan, economic evaluation was institutionalized in the pricing system for drugs and medical devices in 2019. Research methods and policy applications of economic evaluation are expected to progress. However, these methods have not been sufficiently implemented regarding children. This study aims to review how pediatric economic evaluation has been conducted and identify issues regarding its implementation and utilization in Japan. PubMed, Ichushi Web, the National Health Service Economic Evaluation Database, and the Pediatric Economic Database Evaluation database were used to search for literature on complete pediatric economic evaluations in the Japanese healthcare system. Fifty-six journal articles were included in this study, of which more than half were evaluations of vaccines. Cost-utility analysis was the most common method used (41.1%), however, none of these studies measured quality of life values in Japanese children using preference-based scales. There have been few pediatric economic evaluations in Japan, and the disease categories mentioned are limited. It is necessary to conduct economic evaluations and demonstrate the value of pediatric care to society. This will help secure medical resources to improve the health of children, despite there being numerous methodological issues in pediatric economic evaluations.
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Original Article
Title
Intracranial Hemorrhage in Infancy Due to Late-onset Vitamin K Deficiency Bleeding Disorder with Comorbid Cholestatic Diseases: a Hospital-based Study in Japan
Author
Akiko Sasaki1) Hiroo Yamamoto2) Itaru Hayakawa1) Hiroshi Hataya2) Akira Ishiguro3)4) and Yuichi Abe1)
1)Neurology, National Center for Child Health and Development
2)General Pediatrics, Tokyo Metropolitan Children's Medical Center
3)Center for Postgraduate Education and Training, National Center for Child Health and Development
4)Hematology, National Center for Child Health and Development
Abstract
[Background] Vitamin K (VK) prophylaxis to prevent vitamin K deficiency bleeding (VKDB) in infants vary between countries. In Japan, the nationwide campaigns to promote oral VK supplementation since 1983 has led to a significant decrease in incidences of VKDB. However, intracranial hemorrhage (ICH) due to VKDB in infants with biliary atresia albeit oral VK is common. Clinical characteristics of ICH due to VKDB after nationwide oral VK campaigns is not fully elucidated.
[Methods] A multicenter hospital-based retrospective study gathered all the ICH cases due to VKDB between 2010 and 2020.
[Results] Seven ICH cases due to VKDB were found. All harbored comorbid cholestatic diseases. Four received oral VK, while three had no data. In retrospect, the cholestatic disease could have been diagnosed before ICH in 3 out of 7 patients.
[Discussion] We demonstrated that, after 2010, ICH due to VKDB occurred predominantly in those with comorbid cholestatic liver conditions such as biliary atresia. ICH occurred even after oral VK was given according to guideline-recommended doses and timing.
[Conclusion] VKDB prevention strategies in either general population or those with comorbid cholestatic diseases are under investigation. Our results provide useful information regarding optimal preventive strategies to further reduce the incidence of VKDB in infants.
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Original Article
Title
Psychosocial Factors Associated to Refraining from Seeking Medical Care for Children during the State of Emergency: National Survey of Children's Quality of Life and Health in Japan during the COVID-19 Pandemic
Author
Kenta Imai1)2) Makiko Sampei2) Aurelie Piedvache2) Yusuke Okubo2)4) Naomi Sawada2)4) Mariko Hosozawa5)6) Yui Yamaoka3) Kyoko Tanaka7) Akira Ishiguro1) Mayumi Hangai2)8) and Naho Morisaki2)
1)Center for Postgraduate Education and Training, National Center for Child Health and Development
2)Department of Social Medicine, National Center for Child Health and Development
3)Department of Global Health Promotion, Tokyo Medical and Dental University
4)The School of Public Health, The University of Tokyo
5)Institute for Global Health Policy Research, National Center for Global Health and Medicine
6)Department of Pediatrics, Juntendo University Faculty of Medicine
7)Department of Psychosocial Medicine, National Center for Child Health and Development
8)Department of Pediatrics, The University of Tokyo
Abstract
We investigated the association between parents' socio-psychosocial factors and their refrainment from seeking medical care for their children during the state of emergency declared due to COVID-19 in April 2020, using a nationwide web-based survey conducted among parents with children aged ≤17 years in April-May 2020. We found that higher levels of psychological distress and being a single parent were related to refrainment from both scheduled and unscheduled appointments for parents of preschool children, while higher levels of psychological distress and a poor subjective financial condition were related to refrainment from unscheduled appointments for parents of school children.
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Original Article
Title
Current Situation and Problems Concerning Interfacility Transport of Critically Ill Pediatric Patients in Provincial Areas
Author
Yasushi Saito1) Masahiro Watanabe1) Kazufumi Yaginuma1) Osamu Saito2)3) Takehiro Niitsu2)4) Naoki Shimizu2)3)5) Kazuhide Suyama1) and Mitsuaki Hosoya1)
1)Department of Pediatrics, Fukushima Medical University
2)Fukushima Medical Center for Children and Women, Fukushima Medical University
3)Department of Pediatric Emergency and Critical Care Medicine, Tokyo Metropolitan Children's Medical Center
4)Department of Pediatric Intensive Care, Saitama Children's Medical Center
5)Department of Pediatrics, St. Marianna University School of Medicine
Abstract
With the increasing number of critically ill pediatric patients in the PICU, interfacility transport safety has become a problem. We investigated the actual situation and problems arising from interfacility transport in Fukushima prefecture to provide a basis for constructing a safer transport system.
We included 120 critically ill pediatric patients who had been transported to our hospital between April 2017 and March 2020. We defined adverse events as those that required urgent intervention during or immediately after transport. The patients were classified into the 'adverse event' and 'non-adverse event' groups, and compared regarding factors such as transport method.
The median age was 13 months. Transport methods were ambulance and helicopter in 109 and 11 patients, respectively. Airway management was natural in 96 patients and artificial in 24 patients. The adverse event group had 24 patients, among whom artificial airway was required in 13, which was significantly more frequent than in the non-adverse event group. The total number of adverse events was 32; hypoventilation was most common (eight patients), for which a capnometer was not used.
In order to ensure safe and smooth transport of critically ill pediatric patients, sharing information on monitoring and devices among facilities is essential. Additionally, providing training and knowledge-sharing opportunities is necessary among facilities in each region.
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Case Report
Title
Successful Management of Twin Pregnancy with Maternal Aortic Stenosis
Author
Aiko Isobe1) Takayuki Oyanagi1) Shohei Seno1) Kazuki Yamamoto1) Naofumi Sumitomo1) Kazuki Kodo1) Mariko Hida1) Hiroyuki Yamagishi1) Takao Takahashi1) Daigo Ochiai2) and Mamoru Tanaka2)
1)Department of Pediatrics, Keio University School of Medicine
2)Department of Obstetrics and Gynecology, Keio University School of Medicine
Abstract
Pregnancies with severe maternal aortic stenosis (AS) significantly increases the risk of maternal mortality and intrauterine growth retardation. There are no proper perinatal management guidelines for multiple pregnancies with cardiac disease. Case: A 24-year-old woman with moderate AS since childhood visited our hospital for twin pregnancy. At 20 weeks of gestation, cardiac ultrasound revealed severe AS with maximum flow velocity of 4.6 m/s and maximum pressure gradient of 85 mmHg without heart failure. She was admitted at 23 weeks of gestation for careful perinatal management. We decided to perform a caesarean section at 28 weeks of gestation as an optimal timing for termination. Although long gestation would be preferred for fetus development and neurological prognosis, the risk of maternal heart failure will rise due to increased circulating blood volume. Our main aim was to reduce the risk of maternal mortality as much as possible and also to avoid an urgent termination of two low body weight fetuses. After the elective delivery, maternal AS improved to moderate AS and both babies survived without major complications. Transitional care of patients from childhood is an important issue for pediatricians.
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Case Report
Title
A Case of Vici Syndrome with Recurrent Fever and Myositis
Author
Mayumi Sakaguchi1) Chisaki Moriwaki1) Sachi Uyama1) Hirokazu Tsuchie1) Shinji Hagimoto1) Hiroki Kuranobu1) Masami Togawa1) Tomotsune Doumoto1) Akiko Tamura1) Yasushi Utsunomiya1) Mariko Ookubo2) and Ichizo Nishino2)
1)Tottori Prefectural Central Hospital
2)Department of Neuromuscular Research, National Institute of Neuroscience, National Center of Neurology and Psychiatry
Abstract
Vici syndrome is a rare disorder characterized by unique facial features and multiple organ abnormalities. Average life span is 42 months; common causes of death include respiratory failure following an infection and cardiac failure due to cardiomyopathy. Here, we report a patient with Vici syndrome presenting with congenital albinism and callosal agenesis, complicated by suckling disorder, and dilated cardiomyopathy. At two months of age, the patient began experiencing recurrent fevers of unknown origin. Muscle biopsy performed at three months of age and contrast-enhanced magnetic resonance imaging (MRI), indicated inflammatory myopathy as a possible cause of the recurrent fevers. At one year seven months of age, the patient suffered a fatal sudden exacerbation of respiratory distress following an episode of emesis. Since patients with Vici syndrome have a short average life span, it is important to consider the presence of this disorder based on characteristic clinical features, and to perform the appropriate tests. Furthermore, patients with Vici syndrome rarely experience recurrent fevers due to causes other than infection, but they can be caused by myositis, as we observed in this patient. It is crucial to consider performing muscle biopsy when the symptom of myositis occurs with Vici syndrome, because the histopathological findings may be useful for treatment.
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Case Report
Title
Post Intensive Care Syndrome in a Girl Mimicking Hepatic Encephalopathy
Author
Hiroyuki Takao1)2) Hiro Nakao1)2) Tomomi Fujimura1)2) Akiko Sasaki3) Kyoko Tanaka4) Reiko Ito2) Mitsuru Kubota2) and Akira Ishiguro1)
1)Center for Postgraduate Education and Training, National Center for Child Health and Development (NCCHD)
2)Department of General Pediatrics & Interdisciplinary Medicine, NCCHD
3)Division of Neurology, NCCHD
4)Division of Psychosocial Medicine, NCCHD
Abstract
Post intensive care syndrome (PICS) is a complex symptom mimicking various physical, cognitive, and mental health disorders that occurs in intensive care settings. PICS patients may have prolonged functional deficits and impaired quality of life. We report a case of pediatric PICS which needed to be distinguished from hepatic encephalopathy.
The patient was a 7-year-old girl who developed fulminant hepatitis. Her electroencephalogram showed slow waves and she had grade II hepatic encephalopathy. We started to treat her with plasmapheresis. Though her levels of plasma ammonia and serum hepatic enzymes were decreased, she presented with symptoms of prolonged impaired orientation, executive dysfunction, and systemic muscle weakness. We suspected these symptoms to be related to either PICS or hepatic encephalopathy. Eventually, we diagnosed her condition as PICS because blood levels of ammonia and total bilirubin were continuously normal and her slow-brain waves improved with treatment. The case also fulfilled a criterion of ICU-acquired weakness. We initiated physical and occupational rehabilitation therapies at an early phase. Owing to comprehensive support by various medical professionals, her cognitive and mental health status recovered.
This case suggests the importance of suspecting PICS in patients with neurocognitive symptoms after intensive care. Early rehabilitation and total care by multiple professionals might help social reintegration.
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Case Report
Title
A Case of Compartment Syndrome after Inactivated Influenza Vaccination
Author
Midori Tanikawa1) Hiroki Sakurai1) Hidetsugu Tsukui2) Yuka Asano2) Takehiko Sanada2) and Hiroaki Umebayashi1)
1)Department of Rheumatics and Infectious Disease, Miyagi Children's Hospital
2)Department of Plastic Surgery, Miyagi Children's Hospital
Abstract
We encountered a case of compartment syndrome after vaccination with the 2020/2021 inactivated influenza vaccine. The patient was a 2-year-old boy with no remarkable medical history and no allergic predisposition. In the previous year, he had received the influenza vaccine of the same manufacturer without any serious adverse reaction. He was vaccinated on the posterior aspect of his upper right arm and developed a fever that night. The redness and swelling around the inoculation site expanded over time and eventually reached the chest wall. He underwent fasciotomy on the second day of illness because he developed symptoms suggestive of peripheral neuropathy. The adipose tissue and triceps brachii muscle just below the inoculation site were macroscopically degenerated. Pathological examination revealed necrotic tissues and eosinophils. All culture tests from the lesion site were negative; no bacterial infection was in play.
Given that the redness and swelling were centered on the vaccination site and that macroscopic tissue degeneration was most prominent directly under that site, it is probable that the series of events was attributable to the vaccine.
Influenza vaccination is highly recommended, as it is both safe and efficacious. However, compartment syndrome can be a serious local reaction; prompt and appropriate clinical decisions are required to avoid sequelae.
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Case Report
Title
A Girl with Antibodies Detected in Cord Blood after Maternal COVID-19 Vaccination
Author
Tomoya Takiguchi1) Fujio Kakuya1) Yuki Sasaki1) Hitoshi Okubo1) Hiroaki Fujiyasu1) Masashi Ishikawa2) Yasuhiko Hori2) Dai Kondo3) and Takahiro Kinebuchi4)
1)Furano Kyokai Hospital, Pediatrics
2)Furano Kyokai Hospital, Gynecology
3)Furano Kyokai Hospital, Pharmacy
4)Furano Kyokai Hospital, Laboratory of Medicine
Abstract
Neonates have a higher risk for severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) infection than older children. But maternal antibodies transferred to the neonates could prevent morbidity.
A female neonate was born at 39 weeks and 6 days of gestation to a mother injected with the first dose of the SARS-CoV-2 messenger RNA vaccine at 36 weeks and 6 days of gestation. Because the timing of the vaccination coincided with the birth of the baby, the mother's second dose of the vaccination was given after the birth. The anti-SARS-CoV-2 IgG antibody titer (positive at >1.00 U/mL) in maternal blood and cord blood was 6.66 U/mL and 1.33 U/mL, respectively. Although the mother was a medical worker, she did not have any contact with patients infected with SARS-CoV-2. On the day of delivery, her SARS-CoV-2 polymerase chain reaction test was negative. Therefore, it can be suggested that SARS-CoV-2 vaccination can help develop maternal antibodies, which may transfer to the fetus via the placenta.
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Case Report
Title
Chronic Granulomatous Disease Diagnosed on Immunodeficiency Screenings for Very Early Onset Inflammatory Bowel Disease without Recurrent Infections
Author
Yoshikazu Miura1) Shin-ichiro Hagiwara1) Yusuke Matsuda4) Yuiko Hasegawa2) Kimitoshi Goto3) Kohei Higuchi3) Tamaki Wada1) Takatoshi Maeyama1) Masami Inoue3) Nobuhiko Okamoto2) Taizo Wada4) and Yuri Etani1)
1)Department of Gastroenterology, Nutrition and Endocrinology, Osaka Women's and Children's Hospital
2)Department of Medical Genetics, Osaka Women's and Children's Hospital
3)Department of Hematology/Oncology, Osaka Women's and Children's Hospital
4)Department of Pediatrics, School of Medicine, Institute of Medical, Pharmaceutical and Health Sciences, Kanazawa University
Abstract
Chronic granulomatous disease (CGD), a primary immunodeficiency (PID), is caused by phagocytic dysfunction. In some patients, gastrointestinal manifestations precede CGD diagnosis. Herein, we report the case of a 2-year-old boy with CGD diagnosed during screenings for very early-onset inflammatory bowel disease (VEO-IBD). The patient had no history of recurrent infections and showed growth failure and diarrhea at the age of 4 months. Based on colonoscopy at the age of 2 years, he was diagnosed with VEO-IBD. In the screening tests for VEO-IBD, an auto-oxidative dysfunction of the neutrophil bactericidal activity was detected. Flow cytometry analysis showed the absence of gp91phox, and a genetic test revealed a hemizygous deletion of Xp21.1, including the CYBB gene. Finally, he was diagnosed with CGD. After his gastrointestinal symptoms were improved by corticosteroid and 5-aminosalicylic acid treatment, he underwent umbilical cord blood transplantation as a curative therapy. Endoscopic and pathological findings after transplantation were dramatically improved. Although VEO-IBD is more severe and refractory than elderly-onset IBD, PID screenings may detect VEO-IBD early, which can be cured, and improve its prognosis.
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