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THE JOURNAL OF THE JAPAN PEDIATRIC SOCIETY

Vol.119, No.3, March 2015


Review
1. Cumulative Vaccination Rate of Rotavirus Vaccines in Fuchu City, Tokyo
Original Article
1. Clinical Features of Ovotesticular Disorder of Sex Development
2. Effectiveness of Haemophilus influenzae type b Vaccine and 7 valent Pneumococcal Vaccine on Bacteremia in an Outpatient Pediatric Clinic during 11 Years
3. Effectiveness and Safety of Sequential Oral Antibiotic Therapy for Acute Osteomyelitis in Children
Case Report
1. A Case of Infantile Vitamin B12 Deficiency Caused by Maternal Chronic Atrophic Gastritis
2. A Japanese Boy with Glutaric Acidemia Type 1: Possible Limitation of Acylcarnitine Analysis Alone during the Acute Phase
3. Two Cases of Type 1 Laryngotracheoesophageal Cleft Presenting as Recurrent Wheezing in Infancy


Review
Title
Cumulative Vaccination Rate of Rotavirus Vaccines in Fuchu City, Tokyo
Author
Mana Inoue1) Sayaka Takanashi1) Hiroshi Ushijima1)2) Shoko Okitsu1)2) Hiroshi Sakiyama3) and Masashi Mizuguchi1)
1)Department of Developmental Medical Sciences, Graduate School of Medicine, The University of Tokyo
2)Division of Microbiology, Department of Pathology and Microbiology, Nihon University
3)Sakiyama Children's Clinic
Abstract
In Japan, rotavirus vaccines, namely Rotarix and RotaTeq, were introduced in November 2011 and July 2012, respectively. Their vaccination rates, however, have not been reported by local governments as they are currently in the category of optional vaccines.
We studied the cumulative vaccine coverage rates using vaccine records in the mother and child health handbooks of children at 18-month well-child visits in Fuchu-city, Tokyo, Japan in September and October 2013.
The vaccination rate of Rotarix or RotaTeq was 33.4% (95%CI: ±4.6%). Most children were vaccinated in the recommended period. The number of children vaccinated was significantly smaller with Rotarix or RotaTeq than with universal vaccines. Moreover, significantly fewer children were vaccinated against rotavirus than against Hib and varicella. The lack of financial support and disease recognition may have resulted in the low vaccination rate of rotavirus vaccines. Rotavirus vaccines should be included as universal vaccines in accordance with the recommendation of WHO for every nation.




Original Article
Title
Clinical Features of Ovotesticular Disorder of Sex Development
Author
Mari Hasegawa1) Shinobu Ida1) Kenji Shimada2) Yasuko Shoji1) Kei Matayoshi1) Akiko Konishi1) Hiroyuki Yamada1) Yuri Etani1) and Megumi Kosugi3)
1)Department of Gastroenterology and Endocrinology, Osaka medical Center and Research Institute for Maternal and Child Health
2)Department of Urology, Osaka medical Center and Research Institute for Maternal and Child Health
3)Department of Child Psychiatry, Osaka medical Center and Research Institute for Maternal and Child Health
Abstract
We retrospectively evaluated the clinical features of 12 patients with ovotesticular disorder of sex development who were treated at our hospital between 1988 and 2012. Six children are currently being raised as male and 6 as female; the family registration of 1 child was changed from male to female at 4 months of age. The most common karyotype is 46, XX, and the most common type of gonadal distribution was bilateral ovotestis. Nine patients were born with a uterus. The appearance of the external genitalia varied. Gonadal biopsies were performed in all patients for definitive diagnosis. Parents were thoroughly informed of the relevant pathophysiology so that they could take part in the gender-assignment process. Six children (2 males and 4 females) are currently at least 14 years of age. Puberty occurred spontaneously in 2 males and in 2 of 4 females with a single ovary. Psychological support for older children is provided by physicians, nurses, and clinical psychotherapists; none of the children currently suffer from gender-identity issues. In our patient population, we found varied appearances of the external and internal genitalia, gonadal position, and histology. There are benefits and drawbacks to choosing either a male or female gender for patients with this disorder. Therefore, it is important for clinicians to gather as much medical information as possible and to consider the pathophysiology carefully, and for parents to take part in the gender-assignment process.




Original Article
Title
Effectiveness of Haemophilus influenzae type b Vaccine and 7 valent Pneumococcal Vaccine on Bacteremia in an Outpatient Pediatric Clinic during 11 Years
Author
Mitsuru Fukazawa
Fukazawa Pediatric Clinic
Abstract
Background and Aim: Hib vaccine and 7 valent pneumococcal vaccines (PCV7) were introduced as routine vaccination from the end of 2010 in Japan. The efficacy of these vaccines was evaluated from the incidence of bacteremia at our clinic.
Methods: We had investigated the bacteremia from 2002 to 2013, and had examined the frequency of bacteremia and the changes of pneumococcal serotypes in three stages. The first stage was from 2002 to 2010 before the introduction of routine vaccination, the 2nd stage was in 2011 during the introduction of vaccination and the 3rd stage was from 2012 to 2013 after the introduction of vaccination.
Results: Incidence of Hib bacteremia with an annual average of 2.1 cases in the first stage decreased to 0 cases, (100% decrease, p< 0.05) in the 2nd and the 3rd stages. Incidence of pneumococcal bacteremia with an annual average of 8.7 cases in the first stage decreased to 4.0 cases (54.2% reduction) in the 2nd stage, and decreased to 2.5 case (71.4% reduction) in the 3rd stage (p< 0.01). The ratios of PCV7 serotypes with 83.3% (45/54) in the first stage decreased to 50.0% (2/4) in the 2nd stage, and decreased to 0.0% (0/5) in the 3rd stage. The ratio of PCV13 serotypes with 92.6% (50/54) in the first stage decreased further to 0.0% (0/4) in the 2nd stage. However the ratio of PCV13 serotypes re-increased to 60% (3/5) in the 3rd stage with the rise of serotype 19A.
Conclusions: Hib and pneumococcal bacteremia had decreased shortly after the introduction of routine vaccination. However, an increase of pneumococcal bacteremia with serotype 19A was observed. Future changes of the incidence of pneumococcal serotypes by the introduction of PCV13 as a routine vaccination deserves attention.




Original Article
Title
Effectiveness and Safety of Sequential Oral Antibiotic Therapy for Acute Osteomyelitis in Children
Author
Yosuke Onoyama1) Kensuke Shoji2) Hirokazu Sakai1) and Isao Miyairi2)
1)Department of General Pediatrics and Interdisciplinary Medicine, National Center for Child Health and Development
2)Division of Infectious Diseases, Department of Medical Subspecialties, National Center for Child Health and Development
Abstract
Prolonged antibiotic therapy is recommended for the treatment of acute osteomyelitis as inadequate therapy is associated with chronic sequelae. Few reports have evaluated the efficacy and safety of sequential oral antibiotic therapy in Japan.
We performed a retrospective chart review of children diagnosed with acute osteomyelitis from 2002 to 2012 at our institution. Twenty eight children with acute osteomyelitis were identified, of whom 24 received sequential oral antibiotics after parenteral therapy. At the time patients were switched to oral therapy, all patients were afebrile, had improvement in symptoms, and had a C-reactive protein level less than 1.0 mg/dl. Treatment was successful in all patients and children who had switched to oral therapy had a significantly shorter duration of hospitalization (16.5 days vs 50.5 days; P=0.001).
We also noted a profound effect of preceding oral antibiotics on blood culture results. While 14 of the 18 children without preceding oral antibiotics had positive blood cultures, none of the 10 children who received preceding antibiotics had positive blood cultures making the etiological diagnosis difficult (P< 0.001). Preceding antibiotic use was associated with a delay in initiation of parental therapy, suggesting that injudicious use of oral antibiotics result in delay of optimal therapy.




Case Report
Title
A Case of Infantile Vitamin B12 Deficiency Caused by Maternal Chronic Atrophic Gastritis
Author
Taihei Hayashi1) Kazuyuki Iwai1) Hideo Tsuda1) and Yosuke Shigematsu2)
1)Department of Pediatrics, Fukui Prefectural Hospital
2)Department of Health Science, Faculty of Medical Sciences, University of Fukui
Abstract
We report a 10-month-old boy who had feeding difficulty and failure to thrive since 4 months of age. He had been exclusively fed with breast milk and had not commenced weaning before being referred to our hospital. On admission, he had hypotonia, developmental delay, and normocytic anemia. The brain MRI showed diffuse brain atrophy. Laboratory findings revealed an increase in serum and urinary methylmalonic acid and urinary homocystine, and a decrease in plasma methionine and serum vitamin B12. After beginning hydroxocobalamin therapy, his clinical symptoms and laboratory findings improved. Because chronic atrophic gastritis and vitamin B12 deficiency were present in his mother, maternal atrophic gastritis appears to have caused the vitamin B12 deficiency in the boy. Re-analysis of his dried blood spots for infantile mass screening revealed an elevated ratio of propionylcarnitine-to-methionine. Therefore, the propionylcarnitine-to-methionine ratio might be useful for the early detection of newborns with vitamin B12 deficiency of maternal origin.




Case Report
Title
A Japanese Boy with Glutaric Acidemia Type 1: Possible Limitation of Acylcarnitine Analysis Alone during the Acute Phase
Author
Kohei Haraguchi1) Tatsuharu Sato1) Sumito Dateki1) Asako Tokizawa1) Toshihiko Shirakawa1) Akiko Nakatomi1) Yuki Hasegawa2) Kenji Yamada2) Hironori Kobayashi2) Seiji Yamaguchi2) and Hiroyuki Moriuchi1)
1)Department of Pediatrics, Nagasaki University Hospital
2)Department of Pediatrics, Shimane University School of Medicine
Abstract
We report a 5-month-old Japanese boy with glutaric acidemia type 1 (GA1), who did not show any abnormality in the blood acylcarnitine profile in the acute stage. He was referred to Nagasaki University Hospital because of acute encephalopathy and extrapyramidal symptoms. Computed tomography demonstrated marked enlargement of the bilateral Sylvian fissures. Although no elevation of glutarylcarnitine (C5-DC) was detected at admission, a diagnosis of GA1 was made by urine organic acid analysis that showed an excess of 3-OH-glutaric acid as well as a large amount of glutaric acid. Acylcarnitine analysis of the dried blood Guthrie card collected in his neonatal period for newborn screening demonstrated an elevated level of C5-DC.
We should recognize that GA1 could be overlooked during the acute phase by acylcarnitine analysis alone, probably due to a profound carnitine deficit. Early diagnosis and intervention is essential for a better outcome in patients with GA1. In this regard, expanded neonatal mass screening by tandem mass spectrometry is promising.




Case Report
Title
Two Cases of Type 1 Laryngotracheoesophageal Cleft Presenting as Recurrent Wheezing in Infancy
Author
Takemasa Nomura and Tetsuya Kibe
Department of Pediatrics, Seirei-Mikatahara General Hospital
Abstract
Laryngotracheoesophageal cleft (LTEC) is a rare congenital anomaly, which arises from an incomplete separation of the esophagus and trachea, resulting in a channel defect between the pharyngoesophagus and laryngotrachea. The diagnosis of type 1 LTEC presents a clinical challenge because of the frequent overlap of symptoms with more common childhood respiratory illnesses like asthma. We report two patients with type 1 LTEC, diagnosed at age 4 year and 11 months old, respectively, with a history of repeated episodes of wheezing which were life-threatening on some occasions. In both cases, treatment for asthma, including systemic steroid administration, and gastroesophageal reflex were not effective. Routine investigations including flexible laryngofiberscopy did not reveal the cause of the episodes. Rigid bronchofiberscopy with palpation of the interarytenoid region under general anesthesia revealed a cleft to the level of the vocal cords in both cases. The 4-year-old patient received endoscopical surgical repair after several months of conservative management. Although episodes of wheezing decreased dramatically, psychological problems, such as anticipatory vomiting anxiety, remained. The younger patient gradually improved after around 1 year of conservative management.
These cases suggest that a diagnosis of type 1 LTEC should be considered in patients with recurrent refractory wheezing and a poor response to bronchodilation, particularly in combination with coughing and/or vomiting related to food intake.




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