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THE JOURNAL OF THE JAPAN PEDIATRIC SOCIETY
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Vol.125, No.1, January 2021
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Original Article
Title
Epidemiology of Respiratory Syncytial Virus Infection and Challenges in Determining an Accurate Palivizumab Implementation Period in Japan
Author
Gaku Murakami and Hiroaki Ito
Department of Pediatrics, Kameda Medical Center
Abstract
Due to the recent changes in the seasonal patterns of respiratory syncytial virus (RSV) infection, the Japanese consensus guidelines for the use of palivizumab recommend an early prediction of RSV epidemics to determine the accurate period for palivizumab implementation. Moreover, the guidelines suggest that the annual commencement of palivizumab administration in each prefecture should be implemented in the same month. However, no study has investigated the difference in epidemicity of RSV in the entire prefecture and each region within the prefecture. Therefore, this study aimed to evaluate these aspects. In this study, Chiba prefecture was divided into four regions, and retrospective epidemiological analyses of the RSV infection was performed based on data obtained from the Infectious Disease Weekly Report from 2012 to 2019; the analysis involved a comparison of the entire prefecture and each region within the prefecture. The method employed by the WHO for defining an epidemic was adopted to determine the epidemics in this study, which has been previously used for infectious diseases such as influenza. Our analyses revealed that the seasonal epidemics in the regional areas did not always correspond with those in the entire prefecture, except in the northwest region, which has the largest pediatric population. In the middle region of Chiba prefecture, where Chiba-city is located, epidemical gaps of >4 weeks were observed on half of the occasions. In conclusion, the palivizumab implementation period should be decided after analyzing the local RSV epidemics more precisely using the data of each region, obtained from the Infectious Disease Weekly Report, together with the local epidemic indexes, such as hospitalization surveillance.
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Original Article
Title
Questionnaire on Challenges in Providing Support for Pediatric Home Care among Subjects in Various Occupations
Author
Seigo Korematsu1)2) Asuka Nagahama3) Mutsumi Akaishi4) Keisuke Sato5) and Koichi Iida4)
1)Nakatsu Municipal Hospital
2)Oita University Faculty of Medicine
3)Sakanoichi Hospital
4)Oita Prefectural Hospital
5)The Holly Mother of Grace Hospital Home for Children and Persons with Severe Motor and Intellectual Disabilities
Abstract
Support for children requiring medical care has been promoted. Workshops for medical personnel are held in various locations. We conducted a survey to understand the needs of participants by occupation.
A questionnaire was conducted among 180 participants who participated in workshops on pediatric home care in Oita prefecture between 2015 to 2019 (16 pediatricians, 12 adult home-care physicians, 4 dentists, 6 pharmacists, 10 pediatric nurses, 34 adult patient nurses, 33 therapists, 6 counseling support specialists, 35 teachers, 3 childcare workers, 10 workers in administrative agencies, 11 guardians).
Common responses included, "knowledge of payment for medical services, etc. (A)" and "knowledge of welfare services (B)"among pediatricians; "A" among adult home-care physicians; "knowledge of severe motor and intellectual disabilities (C)" among pediatric nurses; and "C)" and "B)" among adult patient nurses; "C)" and "cooperation with medical institutions" among therapists; "support in emergencies" among teachers; and "cooperation with other occupations" among guardians.
Different occupations have different challenges. Based on these results, we plan to conduct workshops to promote mutual collaboration for home-care patients and their guardians.
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Case Report
Title
A Case of Hereditary Hemorrhagic Telangiectasia with a de novo RASA1 Mutation
Author
Hiroko Hirai1) Hideyuki Nakaoka1) Keijiro Ibuki1) Sayaka Ozawa1) Takahiro Homma2) Ikuo Hashimoto3) Takashi Okabe4) Fukiko Ichida5) Tadashi Kaname6) and Keiichi Hirono1)
1)Department of Pediatrics, University of Toyama
2)Department of General Thoracic and Cardiovascular Surgery, University of Toyama
3)Department of Pediatrics, Toyama City Hospital
4)Okabe Pediatrics Clinic
5)Department of Pediatrics, International University of Health and Welfare
6)Department of Genome Medicine, National Center for Child Health and Development
Abstract
Most cases of pulmonary arteriovenous fistula (PAVF) are congenital and are caused by a growth dysfunction of the capillary vessels during the perinatal period. While there are some asymptomatic cases, which are detected by chance during medical examinations, most cases are associated with hypoxemia in adulthood and show such symptoms as the presentation of dyspnea, cyanosis, and clubbed fingers.
We herein report a case of a 7-year-old girl with PAVF. She had cyanosis and clubbed fingers. The PAVF was extensive in the right lower lung and difficult to embolize despite attempts at performing percutaneous catheterization. The patient underwent thoracoscopic right lower lobectomy. Her postoperative course was good, and her cyanosis and fatigue consequently improved.
In addition, she had characteristic symptoms, such as skin hemangioma, epistaxis, and conjunctival injection, indicating the onset of hereditary hemorrhagic telangiectasia (HHT). We therefore performed a whole-exome sequencing analysis on her family, which revealed a de novo pathogenic variant (exon10: c.810delA) of RASA1.
The course of the present case differed from the typical course of HHT due to the absence of a family history, young onset of the disease, and the fact that the PAVF was not diffuse. These features may represent a characteristic phenotype of HHT caused by the RASA1 variant.
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Case Report
Title
Unilateral Nephrectomy and Peritoneal Dialysis Followed by Renal Transplantation in Congenital Nephrotic Syndrome of the Finnish Type
Author
Takeshi Nagasawa1) Kenichiro Miura1) Naoto Kaneko1) Tomoo Yabuuchi1) Kiyonobu Ishizuka1) Hiroko Chikamoto1) Yuko Akioka1) Yuji Tomii2) Syuichiro Fujinaga2) Yoshiyuki Otomo2) and Motoshi Hattori1)
1)Department of Pediatric Nephrology, Tokyo Women's Medical University, School of Medicine
2)Department of Nephrology, Saitama Children's Medical Center
Abstract
In patients with congenital nephrotic syndrome of the Finnish type (CNF) who have Fin-major/minor mutations in the NPHS1 gene, one-step bilateral nephrectomy and peritoneal dialysis (PD) are performed in early infancy due to massive proteinuria. In contrast, unilateral nephrectomy in early infancy and the initiation of PD followed by two-step bilateral nephrectomy are performed in patients with other NPHS1-gene mutations who have less severe proteinuria. However, this includes bilateral nephrectomy that requires PD management in anuric infants and children, which can be difficult and complicated. In this report, we describe two cases of CNF patients who underwent unilateral nephrectomy and renal transplantation without bilateral nephrectomy. Both patients were given a diagnosis of CNF caused by NPHS1 mutations other than Fin-major/minor mutations. In both patients, PD was initiated approximately 1 year after unilateral nephrectomy and they became anuric approximately 2 years after the PD initiation. One case had repeated episodes of infection and severe growth retardation after unilateral nephrectomy. Both patients underwent renal transplantation at approximately 5 years of age, and their post-transplant courses were favorable with good graft function and growth improvement.
Thus, unilateral nephrectomy and PD initiation followed by renal transplantation can be a feasible treatment option for CNF. However, two-step bilateral nephrectomy combined with renal transplantation should be considered when severe growth retardation is evident after unilateral nephrectomy.
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Case Report
Title
Two Cases of Skin Hypopigmentation after Influenza Vaccination
Author
Fumihito Nozaki Saeko Sasaki Minoru Shibata Mioko Mori Mariko Ishihara Atsushi Mori Kenji Inoue Takeo Kato and Takashi Kusunoki
Department of Pediatrics, Shiga Medical Center for Children
Abstract
Influenza vaccination is one of the important preventive vaccinations. We report two cases with skin hypopigmentation after influenza vaccination. Patient 1 was a 4-year-old girl. Patient 2 was a 5-year-old girl. Focal inflammatory reactions occurred after influenza vaccination in both patients. After focal reactions improved, hypopigmented skin lesions were evident at the injected side. Functions of the arm on the injected side remained normal. In Patient 1, skin hypopigmentation disappeared 9 months after injection with no treatment, and no recurrence has been seen during 2 years of follow-up. In Patient 2, observation with no treatment remains ongoing. Consent for skin biopsy could not be obtained. Postinflammatory hypopigmentation was hypothesized as the cause, because hypopigmented skin lesions occurred after focal inflammatory reactions. To the best of our knowledge, this represents the first report of skin hypopigmentation after influenza vaccination. No description of skin hypopigmentation is provided in patient package inserts for influenza vaccination. Skin hypopigmentation is a rare side reaction of influenza vaccination that can occur as an adverse reaction to the vaccination.
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Case Report
Title
Inhaled Hypertonic Saline in Siblings with Primary Ciliary Dyskinesia
Author
Shiori Eguchi1) Takeshi Isoda1) Tomoya Kaneda1) Kento Inoue1) Akihiro Hoshino1) Aoi Morishita1) Motoi Yamashita2) Shigeru Takishima3) Masakatsu Yanagimachi1) Masatoshi Takagi1) Hirokazu Kanegane2) Kohsuke Imai4) and Tomohiro Morio1)
1)Department of Pediatrics, Tokyo Medical and Dental University
2)Department of Child Health and Development, Tokyo Medical and Dental University
3)Department of Pediatrics, Soka Municipal Hospital
4)Department of Pediatrics, Perinatal and Maternal Medicine, Tokyo Medical and Dental University
Abstract
Primary ciliary dyskinesia (PCD) is a rare disease characterized by abnormal ciliary ultrastructure and chronic respiratory tract infections without established therapeutic strategies.
Seven- and three-year-old male siblings were referred to our hospital with recurrent upper and lower respiratory tract infections since their early childhood. PCD was diagnosed from electron microscopic observation of nasal mucosa lacking ciliary outer dynein arms and genetic analysis revealing compound heterozygous DNAH5 mutations. Respiratory physiotherapy, inhaled bronchodilators and steroids, expectorants and antibiotics had not prevented recurrent respiratory tract infections. Twice-daily inhalations of hypertonic (10%) saline were introduced when they were twelve and eight years old. During the next two-year observation, episodes of fever above 38°C per month have been reduced significantly in the younger brother (1.042 vs 0.292, p=0.018) and, though nonsignificantly, in the older brother (1.000 vs 0.375, p=0.096).
The efficacy of inhaled hypertonic saline has been reported in cystic fibrosis, which is similar to PCD in that mucociliary clearance is impaired. For PCD, there is only one trial enrolling adult patients. Further studies will be required to determine whether inhaled hypertonic saline prevents respiratory tract infections and improves the quality of life in pediatric PCD patients.
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Case Report
Title
Pediatric MR Enterography of an 8-year-old Boy with Crohn's Disease
Author
Kosuke Tsuchida1) Takahiro Maeda1) Yuta Sasaoka1) Yuhei Kawashima1) Yoshiyuki Sakai1) Nariaki Toita2) and Yukihiko Kawasaki3)
1)Department of Pediatric, Hakodate Municipal Hospital
2)Department of Pediatric, Sapporo-Kosei General Hospital
3)Department of Pediatric, School of Medicine, Sapporo Medical University
Abstract
Crohn's disease often involves small intestinal lesions, and image evaluation of the small intestine is necessary. In western countries, magnetic resonance enterography (MRE), which enables intestinal evaluation using MR imaging, is recommended as a modality that can be repeatedly used without radiation exposure. In Japan, the use of MRE has been given a lot of attention in adults. However, no fixed protocol has been established for MRE in children, and therefore has not been actively implemented. We developed a protocol for pediatric MRE and performed MRE without sedation in 8-year-old pediatric patients with Crohn's disease. MRE confers no radiation exposure, so it is useful for assessing disease activity and treatment effects repeatedly. Moreover, oral administration of a gastrointestinal contrast agent and performing MRE without sedation are possible even in a young child, depending on the preparation and examination method. However, no fixed protocol has been established for pediatric MRE; therefore, the optimal testing protocol must be studied while accumulating cases in the future.
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Case Report
Title
Decision-making Regarding Emergency Surgical Treatment for Pediatric Orbital Floor Fractures
Author
Yuu Haraki1) Izumi Kato1) Satoshi Nakajima1) Mayumi Hara1) Katsuaki Abe1) Masaki Kanazawa1) Yuichiro Otsuka2) Maiko Kubo3) Mariko Kubota4) and Masaru Terai1)
1)Division of Pediatrics, Chiba Kaihin Municipal Hospital
2)Division of Otolaryngology, Chiba Kaihin Municipal Hospital
3)Division of Plastic Surgery, Chiba Kaihin Municipal Hospital
4)Division of Ophthalmology, Chiba Kaihin Municipal Hospital
Abstract
Orbital floor fractures are fractures of the orbital wall that occur when a blunt external force is applied to the orbital region. Entrapment of the extraocular muscle is known to require immediate surgical treatment in order to prevent sequelae such as ocular motility disorders and diplopia. Children with orbital floor fractures can easily have entrapment of the extraocular muscles; therefore, it is of great importance to determine the appropriate timing of surgical treatment.
We report five pediatric cases of orbital floor fractures. Three patients required emergency surgeries. All three patients were transferred to our hospital for symptoms such as difficulty in opening the eye, eye pain, and vomiting, which occurred immediately after eye injury. Remarkable ocular motility disorders and oculocardiac reflex symptoms were observed in all patients. In contrast, two patients who did not require immediate surgical treatment showed neither remarkable ocular motility disorders nor oculocardiac reflex symptoms. It has been reported that the oculocardiac reflex, such as vomiting and bradycardia, suggests entrapment of the extraocular muscles. The three patients who required emergency surgeries also had remarkable ocular motility disorders and oculocardiac reflex symptoms. These symptoms should be used as a reference in determining the indications for emergency surgery.
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Brief Report
Title
Problems in the Diagnosis of Catheter-related Blood Stream Infection in the Pediatric Intensive Care Unit
Author
Hiroki Kato1) Kazuki Kikuyama2) Takanori Funaki3) Kentaro Ide1) Nao Nishimura1) and Satoshi Nakagawa1)
1)Division of Critical Care Medicine, National Center for Child Health and Development
2)Division of Critical Care Medicine, Aichi Children's Health and Medical Center
3)Division of Infectious Disease, National Center for Child Health and Development
Abstract
In the pediatric intensive care unit (PICU), some patients clinically treated for a catheter-related bloodstream infection (CRBSI) do not meet the appropriate diagnostic criteria. Of the 38 patients who were admitted to the PICU at our hospital between May 2012 and May 2015, and with a diagnosis of CRBSI, 17 did not meet the diagnostic criteria. Among these 17 patients, samples could not be collected from 3 patients, while samples from 9 patients did not meet the diagnostic criteria. The problems with CRBSI diagnosis in the PICU were that specimens for blood cultures were difficult to collect from the central venous catheter and that it was difficult to remove the catheter and submit the catheter-tip culture before confirming CRBSI diagnosis.
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