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THE JOURNAL OF THE JAPAN PEDIATRIC SOCIETY

Vol.122, No.10, October 2018


Original Article
1. Natural History and Risk Factors Regarding the Persistence of Isolated Patent Ductus Arteriosus
2. Psychosocial Support for Healthy Growth and Development in Long-term Hospitalized Pediatric Cancer Patients at Okayama University Hospital
3. Assessment of Vitamin D Sufficiency in Breastfed Infants in One Area in Northern Japan
Case Report
1. Diagnosis Difficulty of Isolated Right Ventricular Hypoplasia: Lessons Learned from a Neonate Complicated with Persistent Pulmonary Hypertension of the Newborn
2. A Case of Bacteremia Caused by Nontypeable Haemophilus Influenzae Secondary to Duodenal Perforation Caused by Rotaviral Gastroenteritis
3. A Case of Sweet Syndrome in an Infant with Subglottic Stenosis
4. Three Cases of Gastric Varices Treated with Balloon-occluded Retrograde Transvenous Obliteration in Young Adults


Original Article
Title
Natural History and Risk Factors Regarding the Persistence of Isolated Patent Ductus Arteriosus
Author
Naoko Hamaguchi Sayaka Mii Yasuaki Kishimoto Daichi Fukumi Makoto Oshiro and Tameo Hatano
Department of Pediatrics, Japanese Red Cross Nagoya Daiichi Hospital
Abstract
[Objective] We investigated the relationship between the closure date of ductus arteriosus and gestational age as well as birth weight.
[Patients] 11,063 neonates born between January 2009 and May 2016 were examined by echocardiography, and isolated patent ductus arteriosus (PDA) was observed in 755 babies. Patients with chromosomal aberrations, congenital malformation syndrome, other congenital heart diseases, and those who underwent surgery for PDA were excluded from this study.
[Methods] Echocardiography was performed at 48 hours or later after birth, and patients with isolated PDA were followed at 3 months, 1 year, and at closure. We also investigated the relationship between the closure rate and gestational age as well as birth weight. We used Fisher's exact test for statistical evaluation.
[Results] PDA was observed in 120 babies (1.1%) at three months and 22 babies (0.2%) at one year. Subsequent closure of PDA was observed in half of those 22 babies. Gestational age shorter than 30 weeks was found to be the significant risk factor for the persistence of PDA. In all 11 cases of spontaneous closure after one year, the size of their PDA was under 1 mm. In 5 cases of PDA in which the minimum size was over 1 mm spontaneous closure was not observed, and 3 of those 5 cases underwent surgery for closure.
[Conclusion] Preterm baby was the risk factor for the persistence of PDA at 1 year of age. We have to develop the treatment strategy for PDA since half of PDA closes at one year old.




Original Article
Title
Psychosocial Support for Healthy Growth and Development in Long-term Hospitalized Pediatric Cancer Patients at Okayama University Hospital
Author
Yasuko Tsurumaru1) Ayumi Okada1)2) Chikako Fujii1) Yoshie Shigeyasu1) Akiko Sugihara1)3) Hisashi Ishida1)4) Kiichiro Kanamitsu1) Kaori Fujiwara1) Kana Washio1) Akira Shimada1)4) and Hirokazu Tsukahara2)
1)Department of Pediatrics, Okayama University Hospital
2)Department of Pediatrics, Okayama University Graduate School of Medicine Dentistry and Pharmaceutical Sciences
3)Kokoro Clinic
4)Department of Pediatric Hematology/Oncology, Okayama University Hospital
Abstract
Recently, the long-term survival rate among pediatric cancer patients has vastly increased; however, in adulthood, psychosocial adaptation problems can arise. Children with malignant diseases often require long-term hospitalization in Japan; therefore, supporting patients psychosocially for healthy growth and development in addition to treating their cancer is important. We therefore examined the effectiveness of a psychosocial support approach and necessary systems for long-term inpatients, especially those with developmental disorders, and their family members.
Of 76 patients admitted for long-term hospitalization (>90 days) between January 2013 and December 2015, 29 who were required to undergo psychosocial intervention according to the department of child psychosomatic medicine were enrolled into our study.
We categorized patients into four groups based on the following consultation reasons for psychosocial evaluation: group 1 (n=10, 34.5%): before bone marrow transplantation; group 2 (n=7, 24.1%): due to developmental assessment results; group 3, (n=5, 17.2%): due to treatment difficulty; and group 4, (n=7, 24.1%): due to family exhaustion.
Developmental disorders were significantly more common in groups 2-4 than in group 1; furthermore, developmental disorders were observed in 9 of 19 patients (47.3%) in groups 2-4. We provided rehabilitation support according to developmental characteristics, supportive interviews and disease education for families, and multidisciplinary cooperation.
When patients rejected their cancer treatment and their family became impoverished, the implemented psychosocial intervention strategies were observed to be effective. Because developmental problems are not identified before hospitalization in many patients, we believe that commencing consultations with family members is a practical approach.




Original Article
Title
Assessment of Vitamin D Sufficiency in Breastfed Infants in One Area in Northern Japan
Author
Kazuhiko Tomimoto1) and Manabu Kinjou2)
1)Tomimoto Children's Clinic
2)Hachinohe City Hospital
Abstract
[Background] Vitamin D deficiency in breastfed infants has recently become a concern due to vitamin D deficiency in pregnant or lactating mothers.
[Objective] To assess vitamin D sufficiency in breastfed infants in the Hachinohe region.
[Methods] Subjects were 155 almost exclusively breastfed infants 3-4 months of age who were seen at this hospital from January-December 2016. Levels of 25-hydroxyvitamin D (25OHD), Ca, P, ALP, and PTH in the blood were examined.
[Results] Of the subjects, 141 (91.0%) were vitamin D-deficient (25OHD levels of 20 ng/mL or lower) and 96 (61.9%) were severely vitamin D-deficient (25OHD level below 12 ng/mL). Vitamin D levels in the summer and fall differed by 11 ng/mL. Levels of 25OHD and PTH correlated. The turning point for the PTH level was a 25OHD level of 9.79 ng/mL (95% CI: 6.65-12.93). Bone changes were noted in fewer than 12 (19.7%) of 61 subjects.
[Discussion] Most infants who were almost exclusively breastfed were vitamin D-deficient, but bone changes were seldom evident. The level of Ca in breast milk was relatively constant. Despite vitamin D deficiency, Ca was absorbed through passive diffusion, so the Ca level in the blood was maintained. The turning point for the PTH level was low with respect to the 25OHD level, and secretion of PTH was inhibited, making bone resorption less likely. This presumably accounts for the lower likelihood of bone changes.
[Conclusion] Most infants who are almost exclusively breastfed in the Hachinohe region are vitamin D-deficient, increasing their risk of rickets.




Case Report
Title
Diagnosis Difficulty of Isolated Right Ventricular Hypoplasia: Lessons Learned from a Neonate Complicated with Persistent Pulmonary Hypertension of the Newborn
Author
Hitoshi Irabu Takeshi Futatani Shuhei Fujita Chihiro Taniguchi Ayako Shimao Hiroyuki Higashiyama Keigo Nishida and Kiyoshi Hatasaki
Department of Pediatrics, Toyama Prefectural Central Hospital
Abstract
Isolated right ventricular hypoplasia (IRVH) is a rare cyanotic congenital heart disease characterized by an underdeveloped right ventricle without obstruction of the inlet or outlet from the right ventricle. We describe a male neonate with IRVH who was delivered after 39weeks of gestation by vacuum extraction due to prolonged fetal bradycardia during induced labor. His Apgar scores were 1 and 3 at one and five minutes, respectively. The amniotic fluid was heavily stained and meconium was aspirated by endotracheal suctioning. He was placed on mechanical ventilation and therapeutic hypothermia, nitric oxide inhalation and catecholamines were started. Although echocardiography showed diminished pulmonary arterial flow and a right-to-left shunt via the foramen ovale, the right ventricle was not dilated as usually seen in persistent pulmonary hypertension of the newborn. Despite remarkable improvement of his chest X-ray by day 3, his oxygenation remained low. Additional treatment by endothelin antagonist and prostacyclin, oxygenation had little effect to improve oxygenation. Repeat echocardiography revealed underdevelopment of trabecular musculature and progressive volume reduction of right ventricle, which eventually lead to the diagnosis of IRVH. It is important to consider IVRH when neonates have intractable pulmonary hypertension with disproportionally small right ventricle.




Case Report
Title
A Case of Bacteremia Caused by Nontypeable Haemophilus Influenzae Secondary to Duodenal Perforation Caused by Rotaviral Gastroenteritis
Author
Michiru Sasaki1) Hidenori Nakagawa1) Tadashi Shinomoto1) Masato Nishihara1) Naoko Imuta2) Junichiro Nishi2) Hiroyuki Sano1) and Makoto Nabetani1)
1)Department of Pediatrics, Yodogawa Christian Hospital
2)Department of Microbiology, Kagoshima University, Graduate School of Medical and Dental Sciences
Abstract
Gastroduodenal perforation and ulceration are complications of rotaviral gastroenteritis. Secondary peritonitis is often caused by microflora implicated in the primary gastrointestinal disease processes. Therefore, peritonitis secondary to gastroduodenal perforation is usually caused by gastroduodenal or oropharyngeal flora derived from swallowing. However, there have been no reports of peritonitis caused by oropharyngeal flora secondary to gastroduodenal perforation in infants. We report a case of 2-year-old girl with bacteremia caused by nontypeable Haemophilus influenzae (NTHi) secondary to duodenal perforation caused by rotaviral gastroenteritis. She presented with vomiting and was diagnosed with rotavirus gastroenteritis. Three days later, she developed fever and altered mental status, and was therefore transferred to our emergency department. Ten hours later, the patient developed peritoneal irritation, and intraabdominal free air was identified on abdominal X-ray; therefore, emergency surgery was performed. During the operation, we diagnosed peritonitis secondary to duodenal perforation. Following the operation, NTHi was isolated from the blood culture. In addition, H. influenzae and α-hemolytic Streptococcus, which are thought to be oropharyngeal flora, were isolated from the ascites culture.




Case Report
Title
A Case of Sweet Syndrome in an Infant with Subglottic Stenosis
Author
Mami Takeoka1) Satoshi Hiraoka2) Takato Nakamoto2) Toyohiro Yamauchi2) Takayo Shoji2) Hajime Katsumata2) Katsuji Karaki2) and Yuji Sekine2)
1)Department of Pediatrics, Mie Prefectural General Medical Center
2)Department of General Medicine, Shizuoka Children's Hospital
Abstract
Sweet syndrome is rare in children. We report a case of Sweet syndrome in an infant with subglottic stenosis. A 1-month-old boy with fever and erythematous skin lesions was first admitted to another hospital for viral upper respiratory infection. Despite treatment, inspiratory stridor developed. Due to progressive respiratory failure, he was transferred to our hospital. He was intubated and supported by mechanical ventilation. Endotracheal intubation was difficult because there was severe stenosis and edema in the subglottis. He underwent tracheostomy for a high degree of subglottic stenosis. We diagnosed Sweet syndrome associated with upper respiratory infection based on clinical presentation and pathological findings of the skin. Erythematous papules improved spontaneously with pigmentation. Tracheomalacia was also found and he went home with cannulation. After the discharge, he had a diagnosis of congenital subglottic stenosis. In this case, we considered that respiratory infection caused exacerbation of congenital subglottic stenosis and contributed to worsened subglottic stenosis. It finally progressed to ventilatory failure. At present, he has no recurrence of Sweet syndrome and his subglottic stenosis is gradually improving.




Case Report
Title
Three Cases of Gastric Varices Treated with Balloon-occluded Retrograde Transvenous Obliteration in Young Adults
Author
Shu Matsushita1)2) Yuki Cho1) Akira Yamamoto2) Yoshiharu Isoura1) Eiji Ehara3) Yosuke Murakami3) Takashi Hamazaki1) and Daisuke Tokuhara1)
1)Department of Pediatrics, Osaka City University Graduate School of Medicine
2)Department of Diagnostic and Interventional Radiology, Osaka City University Graduate School of Medicine
3)Department of Pediatric Cardiology, Osaka City General Hospital
Abstract
We present 3 cases of gastric varices (GVs) treated with balloon-occluded retrograde transvenous obliteration (BRTO). The first case was an 18-year-old man with Wilson disease. He had liver cirrhosis with nodular GV drained by gastrorenal shunt. The varix and the drainer were embolized by BRTO via the shunt. Postoperative endoscopy showed a disappearance of the varix. The second patient was an 18-year-old man who underwent Fontan procedure when he was 4. He had advanced liver fibrosis with a nodular GV drained by gastrorenal shunt. The venous pressur was high under balloon occlusion of the shunt, thus the varix alone was embolized by BRTO. The varix disappeared but worsening of the esophageal varix was detected postoperatively. The last case was a 19-year-old woman who underwent Fontan procedure when she was 2. She had advanced liver fibrosis with a nodular GV drained by gastrorenal shunt. The varix and the drainer were embolized by BRTO via the shunt. The varix disappeared, whereas esophageal varix were exacerbated postoperatively. BRTO is an effective treatment for GVs with gastrorenal shunts in young patients with chronic liver disease, whereas worsening of esophageal varix should be monitored postoperatively.




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