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THE JOURNAL OF THE JAPAN PEDIATRIC SOCIETY
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Vol.118, No.1, January 2014
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Review
Title
The Incidence of Pediatric Bacterial Meningitis before the Availability of Haemophilus influenzae Type B Vaccine and Streptococcus pneumoniae Vaccine (2001 to 2010) in Nagano Prefecture
Author
Nagano Pediatric Clinical Research Group Masashi Utsumi1) Yoshiro Amano1) Fumio Morohashi2) Tsukasa Higuchi3) Hideo Ushiku4) Kuniaki Naganuma5) Tetsuo Nomiyama6) Yuji Inaba7) and Kenichi Koike7)
1)Department of Pediatrics, Nagano Red Cross Hospital
2)Department of Pediatrics, Shinonoi General Hospital
3)Department of General Pediatrics, Nagano Children's Hospital
4)Department of Pediatrics, Saku Central Hospital
5)Department of Pediatrics, Iida Municipal Hospital
6)Department of Preventive Medicine and Public Health, Shinshu University School of Medicine
7)Department of Pediatrics, Shinshu University Hospital
Abstract
The introduction of Haemophilus influenzae type b vaccine and Streptococcus pneumoniae vaccine were delayed in Japan, and they were finally introduced in 2008 and 2010, respectively. The present study aimed to determine the incidence and clinical findings of pediatric bacterial meningitis by analyzing data collected from patients under 15 years of age with bacterial meningitis in Nagano Prefecture between 2001 and 2010, when both vaccines were not widely used. A total of 157 pediatric patients with bacterial meningitis (133 definite cases and 24 probable cases) were enrolled in this study. The incidences of bacterial meningitis in patients under 1 year, under 5 years, and under 15 years of age were 44.6 (95% confidence interval [CI], 31.2-58.0), 14.4 (95% CI, 11.7-17.1), and 5.0 (95% CI, 4.1-5.9) cases per 100,000 population per year, respectively. In 132 patients whose causative pathogens for bacterial meningitis was identified, H. influenzae was predominant (60.6%), followed by S. pneumoniae (19.7%), Streptococcus agalactiae (9.1%), Escherichia coli (4.5%), Listeria monocytogenes (0.8%), and others (6.1%). Nisseria meningitidis was not detected. It was observed that the number of cases of bacterial meningitis caused by H. influenzae and S. pneumoniae began to increase from 2 months of age, and the median ages of the patients at the onset of the 2 infections were 16 months and 9 months, respectively. The findings of the present study may be used as basic epidemiological data to elucidate the efficacy of the Haemophilus influenzae type b vaccine and Streptococcus pneumoniae vaccine in Japan.
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Original Article
Title
Three Children of Inflammatory Myopathy of the Muscle Bundle Interstitium Associated with Systemic Inflammatory Diseases
Author
Masako Kikuchi1) Saori Nagashima1) Satoshi Nozawa1) Taichi Kanetaka1) Toshitaka Kizawa1) Takako Miyamae1) Tomoyuki Imagawa1) Masaaki Mori2) and Shumpei Yokota1)
1)Department of Pediatrics, Yokohama City University School of Medicine
2)Department of Pediatrics, Yokohama City University Medical Center
Abstract
Chronic inflammatory muscle diseases in childhood include juvenile dermatomyositis, polymyositis, and myositis associated with other autoimmune diseases (overlap myositis). We report 3 children who complained of severe muscle pain associated with systemic inflammation and presented with pathologically proven interstitial inflammation between muscle bundles. All of the children visited the hospital with a chief complaint of muscle pain, but not with the skin rash that is characteristic of juvenile dermatomyositis such as Gottron's papules and heliotrope discoloration. Both MRI and electromyography revealed findings suggestive of myositis. Nevertheless, there was no elevation of myogenic enzymes in the blood, while physical examination showed no proximal muscle weakness, despite pain on grasping the muscles. Muscle biopsy specimens of these 3 children were subjected to pathological study, which did not demonstrate degeneration or necrosis of the muscle cells themselves, but revealed inflammatory cells infiltrating the interstitium between muscle bundles that spared the tissue surrounding the blood vessels. This finding was inconsistent with juvenile dermatomyositis, polymyositis, and overlap myositis; therefore, a novel pathological concept of "interstitial myositis" might be needed. Cases 1 and 2 had systemic juvenile idiopathic arthritis as the basis of their illness, while Case 3 was diagnosed with chronic recurrent multifocal osteomyelitis as the underlying disease. Both of these illnesses were considered as systemic inflammatory diseases in which myositis manifested itself as a part of systemic inflammation. We expect similar cases to be identified in the future.
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Original Article
Title
Anaphylaxis Caused by House Dust Mites Present in Food
Author
Yuko Omata1) Naoki Shimojo2) Hisae Kazukawa1) Namiko Ogura1) Hiroko Suzuki1) Akemi Saito3) Kenji Nishioka3) Yuma Fukutomi3) Yuji Kawakami4) and Yoichi Kohno2)
1)Department of Pediatrics, Seikei-kai Chiba Medical Center
2)Department of Pediatrics, Chiba University Graduate School of Medicine
3)Clinical Research Center for Allergy and Rheumatology, Sagamihara National Hospital
4)Laboratory of Environmental Science, FCG Research Institute Inc
Abstract
We report a case of anaphylaxis in a 12-year-old girl caused by the consumption of food that contained mites. The girl had consumed home-made pancakes, made using a pancake mix that was opened before preparation and kept at room temperature for six months. Thirty minutes later, she developed an asthma attack with hoarseness and wheals. Her total serum immunoglobulin E (IgE) level was 1,187 IU/mL. Results of tests for specific IgE antibodies to 2 kinds of house dust mites and to 3 kinds of storage mites were positive. In addition, titers of specific IgE antibodies to wheat, gluten and ω-5-gliadin were all < 0.34 IU/mL. On skin prick testing, she developed immediate-type reactions to the pancake mix that was used for making the pancakes; however, no reactions were observed when a new pancake mix was used in the test. We detected 15.277 Dermatophagoides farinae in a 0.5 g sample of the pancake mix, and the amount of Der1 and Der2 dust mite allergens in the pancake mix was 106 μg/g and 17.0 μg/g, respectively. Thus, in cases of anaphylaxis observed after the consumption of food containing flour, it is important to consider the possibility of the presence of mites -in the flour.
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Original Article
Title
Common Variable Immunodeficiency in a Child with Granulomatous Lymphoproliferative Lesions and Dyspnea
Author
Yoshiaki Shikama Akihiko Shimizu Gen Sasaki Eihiko Takahashi and Kunihiko Akagi
Division of Infection, Immunology and Infection, Kanagawa Children's Medical Center
Abstract
We report a girl presenting with dyspnea, who was finally given a diagnosis of common variable immunodeficiency (CVID) with granulomatous lymphoproliferating lesions. Although the diagnosis of CVID was based on recurrent otitis media and hypogammaglobulinemia, the low number of peripheral blood T lymphocytes and decreased neogenesis of T cells suggested impaired cellular immunity. Chest CT showed a mixture of diffuse macular/granular infiltration and interstitial change of the lungs; MRI showed swelling of the hilar/mediastinal and abdominal paraaortic lymph nodes, and an abnormal mass in the left renal hilum. Tests for cytomegalovirus and Pneumocystis jiroveci were negative. The pathological finding of the abdominal mass was lymph nodes with granulomatous lymphoproliferative change, and her pulmonary lesions were considered to be granulomatous-lymphocytic interstitial lung disease (GLILD) associated with CVID. Although steroid therapy was effective, levels of the serum soluble IL-2 receptor began to increase when we decreased the dose of oral prednisolone, so we started cyclosporine A to safely decrease the steroid dose. CVID cases complicated with granulomatous lesions have been reported to have lower number of switched memory B cells, to be more frequently complicated with autoimmune and lymphoproliferating disorders, and to have shorter lifetimes than noncomplicated cases. Considering the poor prognosis, we think that timely stem cell transplantation may be required.
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Original Article
Title
A Case of Haemophilus Influenzae Type b (Hib) Pneumonia and Septicemia Following Administration of Three Doses of Hib Vaccine in Infancy
Author
Ryosuke Tanaka1) Hiroaki Fujiyasu1) Fujio Kakuya1) Fumie Inyaku2) Hiroshi Sakata3) and Naruhiko Ishiwada4)
1)Department of Pediatrics, Furano Kyokai Hospital
2)Inyaku Children's Clinic
3)Department of Pediatrics, Asahikawa Kosei Hospital
4)Department of Pediatrics, Graduate School of Medicine, Chiba University
Abstract
We report a case of Haemophilus influenzae type b (Hib) pneumonia and septicemia that developed 6 months after having received three doses of Hib vaccine in infancy.
A 10-month-old girl had fever and cough, and despite administration of oral antibiotics, her symptoms persisted. Pneumonia developed and she was referred to our hospital. Her peripheral blood culture was positive for Hib. She was given intravenous antibiotics, which resulted in a successful recovery. Before the onset of Hib disease, her anti-polyribosylribitol phosphate antibody concentration in the blood was 0.46 μg/ml, despite a 3-dose priming with Hib vaccine. This concentration was less than the putative concentration of more than 1.0 μg/ml considered to provide long-term protection against Hib disease. However, after recovery from the infection, her antibody concentration elevated to 19.4 μg/ml. Her immunological examinations were also normal, indicating that there were no specific defects in her ability to initiate a normal immune response.
This case report suggests that an initial vaccination may not be sufficient to protect from invasive Hib disease, and a booster dose should therefore be administered as soon as possible after the age of 1 year.
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Original Article
Title
Therapy for Essential Thrombocythemia in a Twelve-year-old Boy
Author
Hiroyuki Kitamura1) Kei Oota1) Yukiteru Tachibana1) and Makoto Kaneda2)
1)Department of Pediatrics, Abashiri Kosei Hospital
2)Department of Pediatrics, Asahikawa Medical University Hospital
Abstract
Essential thrombocythemia is a disease occurs in children much more rarely than in adults, therefore, patient progress and convalescence, as well as treatment strategy has not been clearly established. We encountered a case of a 12-year-old boy with essential thrombocythemia in whom a diagnosis was establishd after he presented with transient visual impairment. We observed the patient's condition with antithrombotic therapy alone. Although no improvement was noted in the platelet count, the symptoms disappeared.
The prognosis of this disease in children is different from that of adult-onset essential thrombocythemia. The incidence of complications is lower in children than in adults, with a lesser chance of progress to myelodysplastic syndrome or acute myeloid leukemia, therefore, we did not administer the treatment that is conventionally used in adults with the same condition.
Thus, establishment of an optimal treatment strategy is needed for essential thrombocythemia in children by accumulating and studying such cases.
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Original Article
Title
Successful Treatment with Vincristine of a Preterm Infant with Intractable Huge Infantile Hepatic Hemangioma Involving Kasabach-Merritt Syndrome
Author
Satoshi Tanaka Sota Iwatani Toshiki Sofue Kazumichi Fujioka Keiko Wada Hitomi Sakai Masami Mizobuchi Seiji Yoshimoto and Hideto Nakao
Department of Neonatology, Hyogo Prefectural Kobe Children's Hospital Perinatal Center
Abstract
The patient was a boy who was born on Day 5 in Week 31 of gestation, weighing 2,145 g. In the fetal phase, a huge liver tumor was suspected. After birth, we diagnosed infantile hepatic hemangioma involving Kasabach-Merritt syndrome (KMS) by laboratory examination and computed tomography findings. The patient did not respond to various previously reported treatments, steroid hormone, interferon-α, and β-blocker; however, vincristine successfully reduced the size of the hemangioma and the KMS was also resolved. A number of therapies have been reported for the treatment of infantile hemangioma, but none has been uniformly effective because of the variation in this disease. To date, there have been only a few reports of vincristine therapy for infantile hemangioma. We consider that vincristine is worth trying to treat patients with intractable infantile hemangioma involving KMS, especially in life-threatening cases.
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Original Article
Title
A Case of Oral Synechia with Severe Restriction in Mouth Opening Treated Successfully in the Early Neonatal Period
Author
Hiroyuki Saito Daisuke Kinoshita Syoko Tamaki Akiko Kawamura Emiko Sakai Tomohiro Iseki Akihiko Kai Syu Maekawa Kiyoaki Sumi and Masashi Shiomi
Department of Pediatrics, Aizenbashi Hospital
Abstract
Oral synechia-the presence of soft tissue or bony adhesions between the maxilla and mandible-is a very rare congenital anomaly usually recognized at birth, and is followed by airway or nutritional compromise. A male infant was born after 39 weeks of gestation and weighing 3,290 g, without any complications during pregnancy. His Apgar scores were 8 and 8 at 1 and 5 min, respectively. We found 7 adhesions in total: 5 from the hard palate to the floor of the oral cavity and 1 on each side from the alveolar part of the maxilla to that of the mandible. The infant had restricted mouth opening; anteriorly, the mouth opening was 10-15 mm, and thus oral synechia was confirmed, and he also developed mild respiratory distress and nutritional compromise. In addition, he had cleft palate and micrognathia. We divided all 7 fibrous cords under local anesthesia 33 h after birth and the respiratory distress improved dramatically. His mouth opening restriction improved gradually after surgery, and he could be successfully fed using a bottle on postnatal day 10. In the present case, early surgical intervention was successful in resolving respiratory distress and nutritional compromise. Appropriate management of the airway and nutritional compromise is important in patients with oral synechia.
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Original Article
Title
A Case of Bochdalek Hernia in a Child with Volvulus of the Stomach
Author
Shinya Murata1) Atsushi Yoden2) Keisuke Inoue2) Hideki Matsumura1) Mitsuru Kashiwagi1) Maki Koh1) Kenichi Okumura1) Keisuke Okasora1) and Hiroshi Tamai2)
1)Department of Pediatrics, Hirakata City Hospital
2)Department of Pediatrics, Osaka Medical College
Abstract
We present a 3-year-old boy who visited the pediatric department of our hospital complaining of abdominal pain and vomiting. Plain abdominal radiography revealed a hugely distended stomach. An upper gastrointestinal series revealed rotation of the stomach with the pylorus portion displaced over the cardia. Abdominal computed tomography (CT) revealed the stomach to be in the left pleural cavity. Therefore, we diagnosed gastric volvulus and Bochdalek hernia. Although the gastric volvulus was resolved by decompression with a gastric tube, it subsequently recurred. Endoscopic decompression and reduction of the volvulus proved successful, after which the diaphragmatic hernia was repaired and gastropexy was performed.
Gastric volvulus is an uncommon disease, with a relatively high mortality rate (6.7%). Nonoperative mortality is 80%. It should be thought of during differential diagnosis when abnormal dilatation of the stomach bubble is visualized on abdominal X-ray. All pediatricians should be able to recognize acute gastric volvulus itself and master the requisite primary care in emergency settings.
Gastric volvulus is often associated with deformities of adjacent organs, including abnormalities of the diaphragm, disorders of gastric ligaments, asplenia, and malrotation of the intestine. Therefore, examinations for such deformities are extremely important.
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