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THE JOURNAL OF THE JAPAN PEDIATRIC SOCIETY

Vol.119, No.7, July 2015


Original Article
1. Impact of a Subsidized Rotavirus Vaccination Program in the Great East Japan Disaster-affected Area
Case Report
1. Two Cases of Childhood Onset Possible Neurosarcoidosis
2. Bromoderma Tuberosum due to Potassium Bromide Intake
3. When Do MRI Abnormalities Appear in Patients in Acute Encephalopathy with Biphasic Seizures and Late Reduced Diffusion?
4. A Case of Pediatric Histiocytic Necrotizing Lymphadenitis Requiring Differentiation from Hemophagocytic Syndrome Associated with Malignant Lymphoma
5. A Case of Lumbar Hernia Associated with Lumbocostovertebral Syndrome
6. Two Cases of Invasive Pneumococcal Disease after Immunization with the 7-valent Pneumococcal Conjugate Vaccine
7. Endoscopic Cystogastrostomy for a Pancreatic Pseudocyst in a Child with Leukemia


Original Article
Title
Impact of a Subsidized Rotavirus Vaccination Program in the Great East Japan Disaster-affected Area
Author
Toru Fuchimukai1) Tomoharu Oki2) Ken Ishikawa3) Shoich Chida3) Yoshitaka Miura4) Hakuyo Ebara5) Osuke Iwata6) Toyojiro Matsuishi6) Kazuko Wada7) and Yasuhide Nakamura8)
1)Department of Pediatrics, Iwate Prefectual Ofunato Hospital
2)Department of Pediatrics, Iwate Prefectual Takata Hospital
3)Department of Pediatrics, Iwate Medical University
4)Miura Pediatric Clinic
5)Ebara Children's Clinic
6)Department of Pediatrics, Kurume University
7)Department of Pediatirics, Osaka University
8)Graduate School of Human Science, Osaka University
Abstract
Background: After the Great East Japan Earthquake of March, 2011, bold interventions were required to maintain the child healthcare system in disaster-affected areas. Purpose: This study investigated the effectiveness of a subsidized rotavirus vaccination program implemented in the Kesen area, Iwate between January 1, 2012 and March 31, 2014. Methods: The indices used to determine the effectiveness were the number of children hospitalized for rotavirus gastroenteritis (RVGE) and the number of children who visited emergency rooms for gastroenteritis (GE) per 10,000 children aged < 5 years. The study was conducted between January 1, 2009 and December 31, 2013. Results: (1) The number of children vaccinated and the vaccination rate were 367 children (92.4%) in 2012 and 342 children (95.6%) in 2013 in the Kesen area. (2) The number of children hospitalized for RVGE fell by 41% in 2012 and 84% in 2013. In 2013, the number of children hospitalized for RVGE in the Kesen area was significantly lower than that in the 3 regions where the program was not implemented (p< 0.001). (3) The number of emergency patients increased after the disaster struck, but the number of GE patients was significantly lower (2013, p=0.008). Conclusion: The results of this study strongly suggest that the subsidized rotavirus vaccination program was effective. This advanced model program implemented in a disaster-affected area will hopefully lead to the revitalization of the target region and greatly contribute to the advancement of child healthcare services in Japan.




Case Report
Title
Two Cases of Childhood Onset Possible Neurosarcoidosis
Author
Takuya Hiraide Tokiko Fukuda Tomoko Matsubayashi Hidetoshi Ishigaki Miki Asahina Tomohide Taguchi Takeshi Miyamoto and Tsutomu Ogata
Department of Pediatrics, Hamamatsu University School of Medicine
Abstract
We report two pediatric cases with granulomatous uveitis, optic neuritis and myelitis. Case 1, a girl aged 6 years and 5 months, developed gait disturbance and bladder and bowel dysfunction. Case 2, a girl aged 7 years and 1 month, initially presented uveitis, and suffered dysfunction of the bladder and bowel later. Both cases had optic neuritis and granulomatous uveitis characterized by keratic precipitates and retinal periphlebitis. In both cases, the T2-weighted MRI of the spinal cord showed a high-intensity longitudinally extensive lesion in which spotty or patchy enhancement appeared on post-contrast T1-weighted images. Even though the lesions were not determined pathologically, these cases, given the multisystem inflammatory conditions, were most likely indicative of sarcoidosis. Sarcoidosis is a systemic disorder characterized by localized non-caseating granulomas. In most sarcoidosis patients, lung and lymph nodes are involved, however, neurosarcoidosis sometimes lacks system involvement other than the neuronal system. The two cases have been received steroids and immunosuppressants. In cases in which immune-related nervous system diseases are suspected, neurosarcoidosis should be considered.




Case Report
Title
Bromoderma Tuberosum due to Potassium Bromide Intake
Author
Hideshi Kawashima Yu Kobayashi Shinichi Magara Noriyuki Akasaka and Jun Tohyama
Department of Child Neurology, Epilepsy Center, Nishi-Niigata Chuo National Hospital
Abstract
Bromoderma tuberosum is a rare drug rash caused by bromide intake. Recently, bromides have again been used in patients with intractable epilepsy during childhood. We report a girl with holoprosencephaly, who showed various forms of bromoderma. She had been treated with potassium bromide since 4 months of age for intractable epilepsy. At the age of 2 years and 4 months, she developed a single small bromoderma tuberosum. After 6 months, the bromoderma evolved into a purulent ulcer and progressively enlarged. Routine blood tests revealed a high inflammatory response and a cutaneous swab obtained from the pustules grew methicillin-resistant Staphylococcus aureus. We diagnosed bacterial infection of bromoderma tuberosum. We halted intake of potassium bromide and treated with appropriate antibiotics, resulting in immediate improvement. At the age of 3 years, her seizures were still refractory to various antiepileptic drugs, and potassium bromide was prescribed again. After 2 months, a purulent ulcer developed and progressively enlarged on her left cheek. Routine blood tests revealed no inflammation and bacterial culture was negative. We diagnosed acute bromoderma without bacterial infection and halted potassium bromide treatment, resulting in gradual improvement. These results underscore the possibility of occurrence of bromoderma during bromide treatment, regardless of administration duration and dosage. Recurrent bromoderma can deteriorate rapidly in comparison with initial decay, therefore we have to pay attention to the symptoms of bromoderma in cases of re-administration of bromide.




Case Report
Title
When Do MRI Abnormalities Appear in Patients in Acute Encephalopathy with Biphasic Seizures and Late Reduced Diffusion?
Author
Tsutomu Takahashi and Masahiro Ihara
Department of Pediatrics, Saiseikai Utsunomiya Hospital
Abstract
We report a 2-year-old girl whose clinical course and radiographic findings were consistent with acute encephalopathy with biphasic seizures and late reduced diffusion (AESD). She was initially diagnosed with complex febrile seizures.
On day 4 of admission before late seizures, magnetic resonance image (MRI) of the brain already showed subtle subcortical diffusion-weighted abnormalities in the white matter of the right occipital region. On day 5 of admission, she had a cluster of complex partial seizures with progressive subcortical diffusion-weighted abnormalities in the right whole hemisphere and the onset of a global developmental regression.
She was eventually given a diagnosis of hemiconvulsion-hemiplegia (HH) syndrome and AESD. Two year later, her developmental delay, although improved, persisted and she continued to be treated with zonisamide monotherapy and rehabilitation.
Progressive subcortical diffusion-weighted abnormalities appear related to the cluster of complex partial seizures, rather than to the initial seizure. MRI on day 3 to 4 of onset is recommended to detect diffusion-weighted abnormalities. Even if the diffusion-weighted abnormalities are subtle, early intervention may contribute to better neurodevelopmental prognosis.




Case Report
Title
A Case of Pediatric Histiocytic Necrotizing Lymphadenitis Requiring Differentiation from Hemophagocytic Syndrome Associated with Malignant Lymphoma
Author
Ryosuke Matsuno Daisuke Toyama Sachio Fujita Maiko Hanamura Hiroki Tsukada Kosuke Akiyama Hirokazu Ikeda and Keiichi Isoyama
Department of Pediatrics, Showa University Fujigaoka Hospital
Abstract
Histiocytic necrotizing lymphadenitis (HNL) is characterized by fever, lymphadenopathy, and leukopenia as well as lymph node pathology which features a wide histiocytic necrotizing lymphadenitis. We report a case of pediatric HNL requiring differentiation from hemophagocytic syndrome (HPS) associated with malignant lymphoma. A 15-year-old male was previously admitted to another hospital for a remittent fever of 1 month's duration. Blood examination revealed pancytopenia and elevated lactate dehydrogenase levels. Contrast-enhanced computed tomography (CT) revealed swollen lymph nodes in the deep cervical region, both supraclavicular fossa and the axilla. Positron emission tomography/CT revealed swollen lymph node in the inferior portion of the left jaw, bilateral cervical region, right axilla and right abdominal region. Bone marrow aspiration revealed macrophage and monocyte hemophagocytosis. HPS associated with malignant lymphoma was suspected. A week after the former hospitalization, the patient was transferred to our hospital. Although his fever had continued, we pursued observation without treatment because his general condition was good. We conducted a cervical biopsy on the seventh day after admission and diagnosed HNL. His fever resolved on the 11th day and he was discharged on the 17th day. Some reports have demonstrated cases of HNL with HPS or HNL with bone marrow finding like HPS. The clinical features of HNL resemble those of HPS, thereby making it difficult to differentiate the diseases in some cases. The prognosis of HPS with HNL may be good, and we are certain that therapy should not be pursued until accurate diagnosis.




Case Report
Title
A Case of Lumbar Hernia Associated with Lumbocostovertebral Syndrome
Author
Shinjiro Horikawa1) Nariaki Miyao2) Yukako Kawasaki1) Masami Makimoto1) Taketoshi Yoshida1) and Yuichi Adachi2)
1)Division of Neonatology, Maternity and Perinatal Care Center, Toyama University Hospital
2)Department of Pediatrics Faculty of Medicine University of Toyama
Abstract
Congenital lumbar hernia (CLH) is rarely seen and the most common clinical entity associated with CLH is lumbocostovertebral syndrome, which has yet to be reported in Japan. We report a female infant presenting with a soft mass above the left lumbar margin, diagnosed as lumbocostovertebral syndrome.
The 40-week-2-day postmenstrual infant was born vaginally, and the mother was examined for asymmetrical fetal growth restriction (FGR) of the fetus observed from 32 weeks postmenstrual age. No malformations were detected by intrauterine ultrasound examination, and the mother's laboratory test demonstrated low protein S activity (33%), which was considered the cause of the FGR. She had been prescribed low-dose aspirin. The mother remained pregnant because the fetus was growing steadily along a -1.5 SD growth curve. After delivery, physical exam and imaging study revealed a left lumbar hernia, multiple malformations of the rib and spine, scoliosis, meningocele, and syringomyelia, leading to a diagnosis of lumbocostovertebral syndrome. She showed no symptoms of respiratory failure or gastrointestinal disturbance, and was discharged on day 18. Without progression of scoliosis, her weight and height steadily increased with neurologically normal development. The meningocele was repaired at 12 months of age. At 13 months of age, the lumbar hernia was repaired laparoscopically.
The lumbar hernia required surgical closure. Operative repair was recommended before 12 months of age because the hernia defect may enlarge, making primary closure difficult. To decide the best surgical method and most appropriate time of the operation, comprehensive judgment about the status of multiple malformations should be considered.




Case Report
Title
Two Cases of Invasive Pneumococcal Disease after Immunization with the 7-valent Pneumococcal Conjugate Vaccine
Author
Manabu Hamamoto Yoichi Kawamura Yusuke Yoshida Aya Tokiwa Kiyotaka Zaha Ryo Honda Nana Sakakibara Takahiro Noguchi Mamoru Honda Takako Asano Hiroshi Matsumoto Hajime Wakamatsu Hiroyuki Kawaguchi and Shigeaki Nonoyama
Department of Pediatrics, National Defense Medical College
Abstract
Although the 7-valent pneumococcal conjugate vaccine (PCV7) has proved to be highly effective against invasive pneumococcal disease (IPD), non-PCV7 serotype infections, which cause significant morbidity and mortality, remain a critical unresolved issue. Two cases of IPD from Japan are presented; both patients had received adequate vaccinations for their age, with a course of three PCV7 doses. Case 1 involved a 10-month-old boy presenting with sepsis; the patient was discharged after several days of intravenous antibiotic therapy, without any complications. Case 2 involved a 1-year-old boy who was found to have meningoencephalitis. Despite receiving adequate intensive care, the patient developed complications, with severe neurological sequelae. The strains isolated from both cases were serotype 22F, which is not included in PCV7.
Since pneumococcal infections caused by non-PCV7 serotypes have increased following the introduction of PCV7 in other countries, additional epidemiologic research into the current status of IPD is needed in Japan.




Case Report
Title
Endoscopic Cystogastrostomy for a Pancreatic Pseudocyst in a Child with Leukemia
Author
Yuki Hagiwara1) Sachi Sakaguchi1) Hiroyuki Tamaichi1) Hiroyuki Yamada1) Junya Fujimura1) Jinkan Sai2) and Toshiaki Shimizu1)
1)Department of Pediatrics, Juntendo University Faculty of Medicine
2)Department of Gastroenterology, Juntendo University Faculty of Medicine
Abstract
Endoscopic cystogastrostomy has been established as a safe and effective procedure for pancreatic pseudocysts in adults. Here, we describe a 6-year-old boy who was treated with ultrasound-guided endoscopic cystogastrostomy for a pancreatic pseudocyst during leukemia chemotherapy. The patient developed asparaginase-induced acute pancreatitis during chemotherapy for acute lymphoblastic leukemia. Subsequently, a pancreatic pseudocyst was identified and found to be increasing in size despite medical treatment. Endoscopic cystogastrostomy was performed 10 weeks after the onset of pancreatitis. The pseudocyst was treated successfully without complications, and the patient was able to resume leukemia chemotherapy soon after the procedure.




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