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THE JOURNAL OF THE JAPAN PEDIATRIC SOCIETY
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Vol.127, No.4, April 2023
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Original Article
Title
Relationships between Developmental Disorders and Allergic Diseases in School-age Children
Author
Tatsuhiro Mizoguchi1)2)3) Shun Morita1)2) Shohei Kawasaki1)2) Yukiko Inada1)2) and Masafumi Zaitsu1)2)
1)Department of Pediatrics, National Hospital Organization Ureshino Medical Center
2)Department of Pediatrics, Faculty of Medicine, Saga University
3)Mizoguchi Kids Clinic
Abstract
Background: The relationships between developmental disorders, such as autism spectrum disorder (ASD) and attention-deficit hyperactivity disorder (ADHD), and allergic diseases in school-age children remain unclear. Understanding the relationship between developmental disorders and allergic diseases and understanding the characteristics of children with both diseases will improve medical care for both diseases.
Objective: To investigate the relationships between developmental disorders and allergic diseases in school-age children.
Subjects and Method: Subjects were 1,136 outpatients between 6 and 11 years old who visited between January 1, 2018 and December 31, 2018. The prevalence of allergic diseases in children with and without developmental disorders were examined using the diagnosis name in the medical fee information.
Results: The prevalence of allergic diseases was significantly higher among children with developmental disorders (59.9%, 82/137) than among children without developmental disorders (31.9%, 319/999). In children with developmental disorders, boys with ADHD showed allergic diseases significantly more frequently than those without ADHD. Multiple logistic regression analysis showed significant associations between ADHD and allergic diseases (adjusted odds ratio [aOR] 3.73), bronchial asthma (aOR 2.99), allergic rhinitis (aOR 3.05), atopic dermatitis (aOR 2.42), and allergic conjunctivitis (aOR 5.28), and also between ASD and atopic dermatitis (aOR 2.24).
Conclusion: School-age children with developmental disorders, particularly ADHD, may be more likely to have allergic diseases.
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Original Article
Title
Trends in Chronic Disease Mortality Rates for Children in Japan over the Past 50 Years
Author
Akinori Moriichi Erika Kuwahara and Narumi Motegi
Division of Specific Pediatric Chronic Diseases, Research Institute, National Center for Child and Development
Abstract
Objective: To clarify changes in mortality due to chronic diseases among Japanese children over the past 50 years.
Methods: The number of deaths due to chronic diseases in childhood was extracted from the causes of death among children under 20 years of age reported in the Vital Statistics. Age-period-cohort (APC) analysis was used to examine age, period, and cohort effects on annual mortality rates.
Results: The overall mortality rate of pediatric chronic diseases consistently decreased in all age groups under 20 years and decreased to about one-fifth of the rate 50 years ago. The results of APC analysis showed that the age effect had a significant impact on the decline in mortality. The accumulation of medical knowledge and advances in medical technology have led to improvement in treatment methods and contributed to the decline in mortality.
Conclusion: Over the past 50 years, the prognosis for pediatric chronic diseases in Japan has improved significantly. The improvement in prognosis means that more children are surviving to adulthood with chronic diseases, and it is important to provide appropriate support for their social participation.
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Case Report
Title
Infected Pulmonary Aneurysm Caused by Corynebacterium sp. after Pulmonary Artery Banding
Author
Mami Nishiyama1)2) Kentaro Tamura1) Mitsuhide Nagaoka1) Satomi Inomata1) Yukako Kawasaki1) Keijiro Ibuki2) Sayaka Ozawa2) Keiichi Hirono2) Akihiko Higashida3) Masaya Aoki3) Naoki Yoshimura3) Hideki Niimi4) and Taketoshi Yoshida1)
1)Division of Neonatology, Maternal and Perinatal Center, Toyama University Hospital
2)Department of Pediatrics, Faculty of Medicine, University of Toyama
3)First Department of Surgery, Faculty of Medicine, University of Toyama
4)Clinical Laboratory Center, Toyama University Hospital
Abstract
Pulmonary artery banding (PAB) is a common palliative surgical technique for congenital heart disease. Pulmonary artery aneurysm is a rare complication of this procedure, but its etiology and risk factors remain unknown. Moreover, treatment guidelines for pulmonary artery aneurysms have not been established. The present study details our experience with an infant who developed an infected pulmonary aneurysm caused by Corynebacterium sp. after PAB. The patient was a female neonate with 21 trisomy who underwent PAB and ligation of ductus arteriosus at 12 days of age due to double outlet right ventricle and patent ductus arteriosus. Although her postoperative course was good, she developed fever and elevated inflammatory response from day 27. Multiple blood cultures detected Corynebacterium sp., whereas echocardiography revealed vasodilation with a vegetation at the beginning of the ductus arteriosus distal to the PAB, for which a diagnosis of infected pulmonary aneurysm was established. After 8 weeks of treatment with vancomycin and rifampicin, blood cultures came back negative and the vegetation disappeared. However, the aneurysm further enlarged, and the patient underwent pulmonary aneurysmectomy and intracardiac repair at 3 months of age. When signs of infection are present after PAB, clinicians should consider the possibility of an infected pulmonary aneurysm distal to the banding. The development of an infected pulmonary aneurysm after PAB could exacerbate the aneurysms due to infection or jet flow, suggesting the need for careful follow-up.
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Case Report
Title
Clinical Features of Non-dystrophic Myotonia with CLCN1 and SCN4A Concomitant Mutations: A Case Report
Author
Kaori Konno Kentaro Fukuda Yukiko Osawa and Toshimasa Obonai
Department of Pediatrics, Tokyo Metropolitan Health and Medical Treatment Corporation Tama-Hokubu Medical Center
Abstract
Non-dystrophic myotonic syndrome, a genetic disorder characterized by skeletal muscle tonicity without muscle degeneration, is caused by genetic mutations in the skeletal muscle-type chloride ion channel (CLCN1) and skeletal muscle-type sodium channel alpha subunit (SCN4A). Although neonatal onset of SCN4A mutations has been reported, reports on developmental delays are rare. Here, we present a case of a boy with a CLCN1 compound heterozygous missense mutation and SCN4A heterozygous missense mutation who presented with developmental delays and multiple complications. His father, who had also been diagnosed with the syndrome, received pharmacotherapy since infancy. Nonetheless, he had growth retardation since the neonatal period due to marked muscle stiffness, was tube fed for a certain period owing to poor feeding, and underwent radical surgery for secondary cystic diaphragmatic hernia. At 2 years and 6 months of age, he did not gain ambulation and had poor speech. Examinations were performed to determine the underlying cause; however, the coexistence of developmental disorders was ruled out. This is an extremely rare case of a child with early onset non-dystrophic myotonic syndrome inherited from his father and presenting with varied and severe symptoms, developmental delay, and multiple hernia complications. Genetic test results suggested that besides duplication of a gene mutation of paternal origin, a gene mutation of maternal origin, which was considered asymptomatic, also contributed to the disease severity. Our results suggest that even if the diagnostic criteria for the syndrome are fulfilled, an appropriate genetic search can provide important insights into the disease pathogenesis.
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Case Report
Title
Two Cases of Selective Hypo-IgG2emia after Hematopoietic Cell Transplantation
Author
Yurika Sakagawa1) Takahiro Tomoda1) Aoi Morishita1) Kento Inoue1) Tsubasa Okano1) Motoi Yamashita1) Takahiro Kamiya1) Tomoko Mizuno1) Takeshi Isoda1) Masakatsu Yanagimachi1) Masatoshi Takagi1) Hirokazu Kanegane2) Kohsuke Imai3) and Tomohiro Morio1)
1)Department of Pediatrics and Developmental Biology, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University
2)Department of Community Pediatrics, Perinatal and Maternal Medicine, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University
3)Department of Community Pediatrics, Perinatal and Maternal Medicine, Tokyo Medical and Dental University
Abstract
More than one year after bone marrow transplantation for primary immunodeficiency, we encountered two cases of selective hypo-IgG2emia despite normalization of serum immunoglobulin levels, no longer requiring immunoglobulin replacement therapy. Both patients had been treated with rituximab for posttransplant complications.
Because serum IgG2 contains antibodies to capsular bacteria such as Streptococcus pneumoniae, there is a high risk of invasive pneumococcal infection in patients with low IgG2 levels. Until now, immunocompetence after hematopoietic cell transplantation (HCT) has been evaluated using serum IgG, IgA, IgM levels, and recovery of immune cell numbers by flow cytometry as indicators, but it has become clear that selective hypo-IgG2emia exists even in patients with normal serum IgG levels. When selective hypo-IgG2emia is observed after HCT, it may be necessary to continue immunoglobulin replacement therapy or take prophylactic antibiotics. Special attention should be paid to patients treated with rituximab.
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Case Report
Title
An Infant with Bilateral Parietal Skull Fractures Attributable to an Accidental Fall from a Baby Sling
Author
Yuki Nishihashi1) Hiromichi Taneichi1) Shintaro Terashita1) Sadashi Horie1) Asami Takasaki1) Shusuke Yamamoto2) Takuya Akai2) and Yuichi Adachi1)
1)Department of Pediatrics, Faculty of Medicine, University of Toyama
2)Department of Neurosurgery, Faculty of Medicine, University of Toyama
Abstract
Multiple skull fractures in infants are suspected to be due to abuse, but they can be caused by a single accidental impact. We herein report a case of an infant with bilateral parietal fractures attributable to an accidental fall from a baby sling. A one-month-old infant fell down onto a concrete ground from a height of 1 meter, when his mother was carrying him in a baby sling. After the fall, he cried, but he did not show any signs of vomiting or convulsion. The baby sling was made for newborns, and a safety belt was required for its use with infants. However, the belt had not been tied at the time of injury. A single abrasion and two subcutaneous hematomas were observed in the center and on both sides of the parietal region, respectively. Head computed tomography revealed parietal skull fractures. We asked the child protection team to become involved in this case. The circumstances described by the mother were consistent with the physical and imaging findings, and the injuries were thus deemed to be due to an unintentional accident. An infant's skull is malleable, and even a short-distance fall can cause skull deformities and fractures. In the present case, we speculated that the bilateral parietal fractures had been caused by a single accidental impact to the parietal area. The patient did not require surgical intervention and has shown no neurological sequelae. The possibility of an accidental single impact should therefore be considered in cases of multiple skull fractures in infants.
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