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THE JOURNAL OF THE JAPAN PEDIATRIC SOCIETY

Vol.116, No.1, January 2012


1. Differential Diagnosis of Pediatric Cases with Chronic Headache and Those with Clinical Characteristics of Headache with Orthostatic Dysregulation
2. Toxic Epidermal Necrolysis in a 3-year-old Boy during the Recovery Period Following Kawasaki Disease
3. Transmission of the Measles Virus Genotype D9 from Philippines
4. Rapid Improvement of Echocardiographic and Electrocardiographic Findings in a 1-year-old Patient with Beriberi Heart
5. A Pediatric Case of Refractory Bronchial Hemorrhage after Fontan Operation Effectively Treated with Bronchial Occlusion Using a Silicone Endobronchial Watanabe Spigot


Original Article
Title
Differential Diagnosis of Pediatric Cases with Chronic Headache and Those with Clinical Characteristics of Headache with Orthostatic Dysregulation
Author
Hideki Shimomura1)2) Tomohiro Kumada1) Takashi Kusunoki1) Tomoko Miyajima1) Nozomi Oda1) Keiko Saito1) and Tatsuya Fujii1)
1)Department of Pediatrics, Shiga Medical Center for Children
2)Department of Pediatrics, Hyogo College of Medicine
Abstract
Headache and orthostatic dysregulation (OD) are common disorders in Japanese children. In a previous study, 50% of children with OD also had chronic headache but the clinical characteristics were not established. This study investigated the prevalence and clinical characteristics of cases of headache with OD in children with chronic headache. We categorized the headaches of 71 patients who consulted our hospital due to chronic headache based on the International Classification of Headache Disorders 2nd edition (ICHD-II). Of 38 patients diagnosed as having primary headache, 28 (39.4%) had migraine, 9 (12.7%) had tension-type headache, and 1 (1.4%) had cluster headache. Of the 28 patients diagnosed as having secondary headache, 23 (32.4%) had headache with OD, and 2 (2.8%) had sinusitis. The characteristics of headache with OD were a pulsating quality, moderate pain intensity, and each affected child demonstrated a particular time when headache developed. These characteristics are similar to those of migraine. Therefore, 7 OD patients with headache fulfilled the migraine criteria of ICHD-II. Treatment for headache with OD was based on the Japanese clinical guidelines for juvenile orthostatic dysregulation version 1. In 13 (65%) of 20 OD patients treated for headache, headache was improved by non-pharmacological treatment, such as physical manipulation, physical training, and adequate water hydration. The remaining patients improved with specific pharmacological treatments for OD. The results of this study show that the prevalence of headache with OD in children with chronic headache is high and most of these patients can become free of headache with conservative, non-pharmacological treatment.




Original Article
Title
Toxic Epidermal Necrolysis in a 3-year-old Boy during the Recovery Period Following Kawasaki Disease
Author
Yuya Tanaka Shingo Abe Kazuko Kasai Yasuo Nakagishi and Mari Miyoshi
Department of Allergy and Clinical Immunology, Hyogo Prefectural Kobe Children's Hospital
Abstract
High-dose immunoglobulin therapy plus antiplatelet agents (flurbiprofen and aspirin) were administered to treat Kawasaki disease in a 3-year-old boy who was admitted to a local hospital. The patient was discharged after the symptoms improved, while being continuously treated with aspirin. Subsequently, fever, eruption, and mucosal impairment occurred. As these symptoms were not improved by the high-dose immunoglobulin therapy to treat the recurrent Kawasaki disease, the patient was transferred to the author's hospital. Based on clinical and pathological findings, toxic epidermal necrolysis was diagnosed. The basic treatment was withholding aspirin, and providing systemic control such as intravenous rehydration and high caloric nutritional supplement, as well as control of infection, pain, and skin condition. Steroid pulse therapy was also performed as a specific treatment for this case, followed by the gradual discontinuation of steroids. As a result, the systemic epidermolysis that had been observed on admission improved, and the patient showed favorable progress and was discharged 47days after admission. Infectious diseases were not seen. The cause of the symptoms was not indicated by the drug lymphocyte stimulation test (DLST) during hospitalization or after discharge.




Original Article
Title
Transmission of the Measles Virus Genotype D9 from Philippines
Author
Kazutoyo Asada1) Ryouji Ichimi1) Kazunobu Ooya1) Hisashi Tanida1) Takaaki Tanaka2) Shigeru Suga1) and Toshiaki Ihara1)
1)Department of Pediatrics, Mie National Hospital
2)Department of Pediatrics, Kawasaki Hospital
Abstract
An index girl who had traveled to the Philippines was given a diagnosis of measles at a hospital in Mie, Japan in August 2010. There were no reported measles case at that time in the area. A 9 month-old girl (case 2) who visited the same hospital the day after the index girl's visit developed measles 8 days later. Her mother (case 3, mother of case 2) acquired measles, but only developed a fever. The measles strains isolated from the index girl and the girl (case 2) were both genotype D9, which is indigenous in the Philippines and the gene arrangements were homologous. Considering this homology, an unusual disease and the latent period of the virus, it is suspected that the girl (case 2) became infected from the index girl. The mother's measles IgG antibody titer just before onset was 5.4 enzyme immune assay (EIA) level which was protective against measles development. In fact she (case 3) did not develop measles rash but fever. The transferred maternal antibody in the girl (case 2) could be too little to have protected her against the virus.




Original Article
Title
Rapid Improvement of Echocardiographic and Electrocardiographic Findings in a 1-year-old Patient with Beriberi Heart
Author
Takanari Fujii1) Akira Hojo1) Toshinori Nakamura1) Junya Iwasaki1) Takaaki Takayanagi3) Shigeru Uemura2) and Kazuo Itabashi1)
1)Department of Pediatrics, Showa University School of Medicine
2)Cardiovascular Center, Showa University Northern Yokohama Hospital
3)Department of Pediatrics, Tokyo Metropolitan Health and Medical Treatment Corporation Ebara Hospital
Abstract
Background: Thiamine deficiency (beriberi) is an acquired error of carbohydrate metabolism, and can be expressed by cardiovascular manifestations (beriberi heart, shoshin beriberi).
Case: A 1-year-old boy was admitted to our hospital suffering from hyponatremia and convulsions. His medical history included a decreased intake of diet and regular massive intake of approximately 1,500-2,000 ml daily of isotonic drink. His main physical findings were tachycardia, diastolic hypotension, and disturbance of consciousness. Laboratory investigation showed lactic acidosis. Brain natriuretic peptide was 1,100 pg/ml. An echocardiogram showed diffuse hypokinesis of the left ventricle, mild tricuspid regurgitation, and pericardial effusion. An electrocardiogram showed diffuse ST-segment change. These findings dramatically improved after intravenous administration of thiamine.
Conclusions: We observed rapid improvement of echocardiographic and electrocardiographic findings after thiamine treatment in our patient. We conclude that it is important to consider beriberi heart as part of the differential diagnosis in pediatric patients with heart failure accompanied by regular intake of isotonic drink and undernutrition.




Original Article
Title
A Pediatric Case of Refractory Bronchial Hemorrhage after Fontan Operation Effectively Treated with Bronchial Occlusion Using a Silicone Endobronchial Watanabe Spigot
Author
Asako Hayashi (Nakata) Yukiko Kawazu Noboru Inamura Ryo Ishii Yuji Hamamichi and Futoshi Kayatani
Department of Pediatric Cardiology, Osaka Medical Center and Research Institute for Maternal and Child Health
Abstract
A 5-year-old girl was admitted following sudden massive hemoptysis. She had previously undergone a Fontan operation for congenital heart disease (pulmonary atresia with intact ventricular septum) at age 3 with a favorable post-operative outcome. We performed several coil embolizations for the detected collateral arteries but massive hemoptysis occurred repeatedly. We therefore performed emergency bronchial occlusion using a silicone Endobronchial Watanabe Spigot (EWS) for the affected areas. By performing both types of operation, we succeeded in saving the patient's life.
This is the first pediatric case of bronchial occlusion using EWS. Although we experienced spigot migration and re-occlusion, we consider that EWS was effective for the refractory bronchial hemorrhage, which could not be controlled by arterial embolizations alone.
We will continue to monitor her condition, paying close attention to any complications arising due to the long-term EWS placement.




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