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거대 간 종괴와 심방 중격 결손을 동반한 Abernethy 기형 2형 1예
이해정,이지현,허준,강이석,이흥재,서연림,유소영,최연호,Lee, Hae-Jeong,Lee, Jee-Hyun,Huh, June,Kang, I-Seok,Lee, Heung-Jae,Suh, Yeon-Lim,Yoo, So-Young,Choe, Yon-Ho 대한소아소화기영양학회 2008 Pediatric gastroenterology, hepatology & nutrition Vol.11 No.1
저자들은 간의 거대 종괴와 심방 중격 결손으로 수술을 받았던 환아에서 복부 CT와 간 조직 검사를 통해 확인된 Abernethy 기형 2형 1예를 경험하였기에 문헌 고찰과 함께 보고하는 바이다. Extrahepatic portosystemic shunts, known as Abernethy malformations, were first reported by John Abernethy in 1793. They are classified into two types: Type I refers to a congenital absence of the portal vein and Type II refers to a shunt involving a side-to-side anastomosis with reduced portal blood flow into the liver parenchyma. This malformation is so rare that less than 100 cases have been reported in the medical literature. We report the case of a 13-month-old boy who had a congenital extrahepatic portocaval shunt with a hypoplastic portal vein. This case was complicated with an atrial septal defect and a large hyperplastic nodule in the liver. The patient was diagnosed with a Type II Abernethy malformation. We planned on surgical occlusion of the extrahepatic portocaval shunt. However, six months later, the patient had a sudden onset of a fever of unknown origin and developed hepatic encephalopathy. Although he underwent a liver transplantation, he died of acute hepatic failure.
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기관지폐이형성증 환아에서 폐고혈압의 임상 경과 및 예후에 관한 분석: 단일 기관 연구
김지현 ( Ji Hyun Kim ),허준 ( June Huh ),강이석 ( I Seok Kang ),이상일 ( Sang Il Lee ),이흥재 ( Heung Jae Lee ),안강모 ( Kang Mo Ahn ) 대한소아알레르기호흡기학회(구 대한소아알레르기 및 호흡기학회) 2008 소아알레르기 및 호흡기학회지 Vol.18 No.3
목적: 최근 기관지폐이형성증 환아가 늘면서 이로 인한 이차성 폐고혈압도 증가할 가능성이 있으나, 이에 대한 연구는 아직 부족하다. 따라서 폐고혈압이 동반된 기관지폐이형성증 환아의 임상 경과와 예후를 알고자 하였다. 방법: 삼성서울병원 신생아중환자실에 입원한 미숙아 중 기관지폐이형성증이 발생한 환아 240명을 대상으로 의무 기록을 후향적으로 분석하였다. 폐고혈압 동반 여부에 따라 두 군으로 나누고 사망률, 이환율 등의 임상 경과를 비교하고, 폐고혈압 발생군에서 예후를 예측할 수 있는 인자가 있는지 분석하였다. 결과: 총 240명의 기관지폐이형성증 환아 중 19명(7.9%)에서 폐고혈압이 발생하였다. 폐고혈압 발생군은 미발생군에 비해 여아와 부당경량아의 비율이 더 높았고, 기관지폐이형성증의 중증도가 더 높았으며, 신생아중환자실에서 인공환기요법을 시행한 기간이 더 길고, 최대 최고흡기압이 더 높았다. 폐고혈압 발생군에서 사망률이 더 높았고, 신생아중환자실 퇴원 이후 더 오랫동안 산소 치료가 필요하였다. 또한 호흡기 질환으로 인한 재입원 횟수가 더 많았으며, 재입원시 소아중환자실에서 치료받거나 인공환기요법을 시행받는 빈도가 더 높았다. 폐고혈압이 동반된 환자에서 사망과 관련된 예후를 예측할 수 있는 인자는 없었다. 결론: 폐고혈압은 기관지폐이형성증의 치명적이고 심각한 합병증으로, 임상적 특성이나 위험 인자로 예측하기 어려우므로, 특히 여아와 출생 체중 500 g 미만, 부당경량아, 장기간 인공환기요법을 받았거나 중증 기관지폐이형성증을 가진 미숙아에서는 폐고혈압의 발생 가능성에 대하여 상기해야 한다. Purpose: The aim of our study was to determine the clinical course and the prognosis of pulmonary hypertension (PHT) with bronchopulmonary dysplasia (BPD). Methods: Two hundred forty infants who were admitted to the neonatal intensive care unit (NICU) of Samsung Medical Center from January 2002 to December 2006 and were diagnosed with BPD were enrolled in this study. We investigated their medical records retrospectively to investigate any difference between BPD with PHT group and BPD without PHT group in clinical characteristics, mortality and morbidity. Results: Nineteen (7.9%) of the 240 patients with BPD developed PHT. The ratio of females to males was 2.8: 1. The severity of BPD, the small birth weight for gestational age, the duration of mechanical ventilation and the maximal peak inspiratory pressure were significantly associated with the development of PHT. (P=0.000, P=0.007 and P=0.000, respectively) The mortality was higher in the BPD with PHT group than in the BPD without PHT group. (P=0.000) BPD with PHT group required longer duration of oxygen therapy and had more rehospitalization for respiratory illness than the others. (P=0.014) More patients were admitted to the pediatric intensive care unit and received mechanical ventilation therapy in the BPD with PHT group. (P=0.001 and P=0.020, respectively) Conclusions: PHT is one of the causes associated with high mortality and morbidity in BPD patients. Therefore, the physician should be alert to the development of PHT in premature infants with severe BPD, especially if the baby is female, less than 500 g of birth weight, small for gestational age, or supported by mechanical ventilation for a prolonged duration. [Pediatr Allergy Respir Dis(Korea) 2008;18: 243-252]
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폐동맥 고혈압과 심부전으로 발현된 폐분리증과 선천성 엽기종이 동반된 1례
김진규 ( Jin Kyu Kim ),김이선 ( I Seon Kim ),강이석 ( I Seok Kang ),안강모 ( Kang Mo Ahn ),이흥재 ( Heung Jae Lee ),김진국 ( Jin Kook Kim ),허준 ( June Huh ) 대한소아알레르기호흡기학회(구 대한소아알레르기 및 호흡기학회) 2009 소아알레르기 및 호흡기학회지 Vol.19 No.4
저자들은 영아기에 폐동맥 고혈압, 심부전으로 발현된 폐분리증과 선천성 엽기종이 동반된 증례를 진단하였고, 이환된 폐엽절제수술 후 폐동맥 고혈압 및 심부전이 호전된 증례를 경험하였기에 보고하는 바이다. Congenital cystic disease of the lung represents pulmonary sequestration, congenital lobar emphysema, bronchogenic cyst and congenital cystic adenomatoid malformation. It has a variety of clinical manifestations from immediate postnatal respiratory distress to heart failure. Pulmonary sequestration sometimes causes heart failure in neonates through a shunt between an anomalous systemic feeding artery and the pulmonary venous system. We hereby report a case with both pulmonary sequestration and congenital lobar emphysema presenting with congestive heart failure and pulmonary hypertension, which improved after lobectomy. [Pediatr Allergy Respir Dis(Korea) 2009;19:434-439]
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CHARGE 증후군 환자에서 호흡기 문제에 관한 분석: 단일 기관 연구
송상미 ( Sangmi Song ),박미란 ( Mi Ran Park ),김지현 ( Jihyun Kim ),최연아 ( Youn Ah Choi ),허준 ( Jinyoung Song ),강이석 ( June Huh ),정만기 ( I Seok Kang ),정한신 ( Man Ki Chung ),손영익 ( Han Sin Jeong ),안강모 ( Young Ik Son 대한천식알레르기학회 2014 Allergy Asthma & Respiratory Disease Vol.2 No.1
Purpose: CHARGE syndrome consists of multiple malformation including coloboma, heart defect, choanal atresia, growth or developmental retardation, genital anomalies, and ear anomalies. The aim of this study was to evaluate the respiratory problems in children with CHARGE syndrome. Methods: Out of 9 patients with CHARGE syndrome, medical records from 8 patients showing respiratory distress or respiratory failure were retrospectively reviewed. We investigated the causes of respiratory problems by physical examination, endoscopy, echocardiogram, computed tomography, rigid bronchoscopy, swallowing test, and 24-hour impedence monitoring. esults: Five patients required endotracheal intubation soon after birth due to bilateral choanal atresia (n=2) and congenital heart diseases (n=3). Three patients were intubated within a month because of surgery for complex heart diseases (n=2) or recurrent apnea (n=1). Tracheostomy was performed in 3 patients who showed primary or secondary subglottic stenosis. Among 8 patients who had aspiration or respiratory distress after feeding, cricopharyngeal incoordination and gastroesophageal reflux disease were found in 7 and 2 children, respectively. One patient died of aspiration during oral feeding. Conclusion: Patients with CHARGE syndrome manifest respiratory distress or failure due to various causes including congenital anomaly in the airway, cardiac anomaly, neurologic or gastrointestinal problems. Therefore, pediatricians should be alert to the respiratory symptoms and signs in CHARGE syndrome and take active intervention from the birth to improve their long-term prognosis. (Allergy Asthma Respir Dis 2014;2:70-74)
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