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Down Syndrome with Transient Myeloproliferative Disorder, Hepatic Fibrosis, and Hemochromatosis.

Jie Yeon LEE ; Hwi Kyu IM ; Hwang Min KIM ; Baek Keun LIM ; Young UH ; Chan Il PARK

Journal of the Korean Society of Neonatology.2003;10(1):83-87.

Transient myeloproliferative disorder (TMD), which may mimic acute leukemia, occurs in neonates with Down syndrome along with hepatic fibrosis. TMD is recognized shortly after birth or in the neonatal period and is characterized by leukocytosis and thrombocytopenia, which resolve spontaneously in four to six weeks. And hepatic fibrosis is characterized by diffuse intralobular sinusoidal fibrosis, extramedullary hematopoiesis and hemochromatosis. A newborn male infant with Down syndrome, atrial septal defect and ventricular septal defect is reported. He showed abnormal myelopoiesis accompanying characteristic hepatic sinusoidal fibrosis. Knowing the cellular mechanism of hepatic fibrosis and its modulation by growth factors, a pathogenetic link between transient myeloproliferative disorder and the development of liver fibrosis in Down syndrome neonates, association of this triad no longer appears to be accidental.
Down Syndrome* ; Fibrosis* ; Heart Septal Defects, Atrial ; Heart Septal Defects, Ventricular ; Hematopoiesis, Extramedullary ; Hemochromatosis* ; Humans ; Infant ; Infant, Newborn ; Intercellular Signaling Peptides and Proteins ; Leukemia ; Leukocytosis ; Liver Cirrhosis ; Male ; Myelopoiesis ; Myeloproliferative Disorders* ; Parturition ; Thrombocytopenia

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A Case of Transient Myeloproliferative Disorder with Down Syndrome, Presented Hepatosplenomegaly on Prenatal Sonography.

Keun Hye LEE ; Beom Su PARK ; Eun Hwan JEONG ; Bo Ra SON ; Mi Kyeong KIM ; Seung Woon LIM ; Hyeon Jin PARK

Journal of the Korean Society of Neonatology.2003;10(1):78-82.

Down syndrome (DS) is associated with a higher incidence of leukemia than general population; the subtype is acute megakaryoblastic leukemia (AMKL) in 50% of cases. DS is also strongly associated with transient myeloproliferative disorder (TMD), which is usually diagnosed during newborns and infants. Due to its difficulty in distinguishing TMD from acute leukemia (AL), the diagnosis of TMD should be made with extreme caution. Unlike AL, most cases of TMD resolve spontaneously within 3 months; blast cells disappear within 8 weeks in 80% and within 10 weeks in 90% of the surviving patients. Some infants with TMD, however, may have a severe complication leading into life-threatening clinical course with hepatosplenomegaly, lymphadenopathy, liver impairment, respiratory distress, anemia, infection and hemorrhage. Rarely, AL can develop after remission of TMD. We report a case of TMD with DS in newly born infant who presented hepatosplenomegaly on prenatal sonography and improved with exchange transfusion, steroid.
Anemia ; Diagnosis ; Down Syndrome* ; Hemorrhage ; Humans ; Incidence ; Infant ; Infant, Newborn ; Leukemia ; Leukemia, Megakaryoblastic, Acute ; Liver ; Lymphatic Diseases ; Myeloproliferative Disorders*

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A Case of Prenatally Detected Congenital Lobar Emphysema.

Keun Hye LEE ; Beom Soo PARK ; Il Un JI ; Jong Myeon HONG ; Il Heon BAE ; Keon Kook LEE

Journal of the Korean Society of Neonatology.2003;10(1):72-77.

Congenital lobar emphysema (CLE) is an overinflation of infantile pulmonary lobe caused by air trapping within the affected area. This is usually diagnosed on radiological findings postnatally. Most cases present in the neonatal period with signs of acute or chronic respiratory distress. There are only a few reports of CLE in the literature with prenatal sonographic feature documented. The routine use of prenatal sonography has helped to find fetal anomalies, such as fetal lung mass, that pose a challenge to early diagnosis, appropriate counselling, postnatal follow-up, and better outcome. A boy with CLE was detected by prenatal sonogram. He had a uniformly echogenic right lung with mediastinal shift to the left which was suspected type III congenital cystic adenomatoid malformation prenatally. He showed no evidence of respiratory distress at birth, but experienced progressive tachypnea and chest retraction. He had right middle lobectomy on the seventh postnatal day and pathologic diagnosis of CLE had been made. We report this rare case with brief review of literature. This is the first domestic case which was detected prenatally and confirmed pathologically after lobectomy.
Cystic Adenomatoid Malformation of Lung, Congenital ; Diagnosis ; Early Diagnosis ; Emphysema* ; Follow-Up Studies ; Humans ; Lung ; Male ; Parturition ; Tachypnea ; Thorax ; Ultrasonography

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Two Cases of Fetus in Fetu Diagnosed with Prenatal Ultrasonography.

Kun Song LEE ; Yoon Hee JEE ; Doo Sun LEE ; Jai Hyang GO ; Young Seok LEE ; Jong Min LEE ; Woo Sung PARK ; Young Pyo CHANG

Journal of the Korean Society of Neonatology.2003;10(1):67-71.

Fetus in fetu is a poorly understood and rare congenital malfomation. This is a rare form of monozygotic twin that asymmetric twin becomes internalized in the other twin thus acting endoparasitically. Fetus in fetu can be distinguised from teratoma, because of vertebral column, skeletal axis, and well-differentiated internal organs. We present the findings in the two cases of fetus in fetu that were diagnosed with prenatal ultrasonography. After birth, we removed fetus-like structures and confirmed by pathologic examination. Fetus-like structures were consisted of vertebral column, extremities, and other well-developed internal organs.
Axis, Cervical Vertebra ; Extremities ; Fetus* ; Humans ; Parturition ; Spine ; Teratoma ; Twins, Monozygotic ; Ultrasonography, Prenatal*

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A Study on the Incidence and Risk factors of Cystic Periventricular Leukomalacia in very Low Birth Weight Infants.

Soo Hyun LEE ; Sung Hye KIM ; Kye Hyang LEE ; Dong Kil YOU ; Suk Joo CHOI ; Jong Hee HWANG ; Chang Won CHOI ; Jae Won SHIM ; Sun Young KO ; Soon Ha YANG ; Yun Sil CHANG ; Won Soon PARK

Journal of the Korean Society of Neonatology.2003;10(1):61-66.

PURPOSE: Our study was carried out to estimate the incidence of cystic periventricular leukomalacia (CPVL) and to identify the risk factors for CPVL. METHODS: The medical records and cranial ultrasound scan were reviewed for 321 infants weighing less than 1, 500 g who lived more than 28 days and admitted to the NICU at Samsung Medical Center from October 1995 to December 2001. A multiple logistic regression was performed to identify which factors were independently associated with CPVL. RESULT: CPVL developed in 19 (5.9%) infants of 1, 188+/-236 g birth weight and 28(+6)+/-2(+4) weeks gestational age. Incidence of CPVL according to birth weight and gestational age were as follows respectively: <750 g 5.3%, 750-999 g 5.5%, 1, 000-1, 249 g 3.9%, 1, 250-1, 499 g 7.9% and <25weeks 8.3%, 25-26weeks 6.7%, 27-28weeks 6.5%, 29-30weeks 2.7%, 31-32weeks 11.1%. The mean day of diagnosis of CPVL was 41+/-33 days. Univriate analysis indicate that two clinical variables, prolonged ventilator duration (CPVL: control, 35+/-64 days vs 17+/-26 days, P=0.0184) and severe intraventricular hemorrhage (IVH) (21% vs 2.7%, P=0.0324), were significant predictors of CPVL. The odds ratio estimate and 95% confidence limits are 1.012 and 1.003 to 1.022, respectively for prolonged ventilator duration; 2.6 and 1.044 to 6.602, respectively for severe IVH. CONCLUSIONS: These data suggest that prolonged ventilator duration and severe IVH increase the risk for development of CPVL.
Birth Weight ; Diagnosis ; Gestational Age ; Hemorrhage ; Humans ; Incidence* ; Infant* ; Infant, Newborn ; Infant, Very Low Birth Weight* ; Leukomalacia, Periventricular* ; Logistic Models ; Medical Records ; Odds Ratio ; Risk Factors* ; Ultrasonography ; Ventilators, Mechanical

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Comparison of Postoperative Apneic Episodes after General and Spinal Anesthesia for Inguinal Herniorrhaphy among very Low Birth Weight Infants.

Yu Jin KIM ; Jae Youn KO ; Jong Hee HWANG ; Chang Won CHOI ; Jae Won SHIM ; Sung Shin KIM ; Yun Sil CHANG ; Won Soon PARK

Journal of the Korean Society of Neonatology.2003;10(1):55-60.

PURPOSE: The objective of this study was to compare the incidences of postoperative apnea in between either spinal or general anesthesia for inguinal herniorrhaphy among very low birth weight (VLBW) infants. METHODS: Retrospective chart review for 28 infants less than 1, 500gram birth weight who underwent inguinal hernia repair at Samsung Medical Center from January 1994 to May 2002 was done. These infants had been already weaned from ventilator before inguinal herniorrhaphy. The subjects were divided into two groups according to the type of anesthesia they received for the hernia repair. RESULTS: There were no statistically significant differences in gestational age, birth weight, corrected gestational age, body weight at the time of operation, duration of mechanical ventilator and duration of oxygen requirements in between groups with general anesthesia (13 cases) and spinal anesthesia (15 cases). The incidences of apnea and requirement of oxygen for 24 hours preoperatively did not show significant differences in between two groups, however, the postoperative incidences of apnea, requirement of oxygen and ventilator care were less in spinal anesthesia group. One infant of general anesthesia group who required postoperative mechanical ventilator eventually ended up with tracheostomy due to ventilator weaning failure. CONCLUSION: This study suggests that the spinal anesthesia for VLBW infants who underwent inguinal herniorrhaphy significantly decreased the incidences of postoperative apnea compared to those infants with general anesthesia.
Anesthesia ; Anesthesia, General ; Anesthesia, Spinal* ; Apnea ; Birth Weight ; Body Weight ; Gestational Age ; Hernia, Inguinal ; Herniorrhaphy* ; Humans ; Incidence ; Infant* ; Infant, Very Low Birth Weight* ; Oxygen ; Retrospective Studies ; Tracheostomy ; Ventilator Weaning ; Ventilators, Mechanical

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The Comparison of Severity according to Preceding Causes of Bronchopulmonary Dysplasia in Very Low Birth Weight Infants.

Sung Hye KIM ; Kye Hyang LEE ; Soo Hyun LEE ; Dong Kil YOU ; Suk Joo CHOI ; Jong Hee HWANG ; Chang Won CHOI ; Jae Won SHIM ; Hye Kyung YOON ; Soon Ha YANG ; Yun Shil CHANG ; Won Soon PARK

Journal of the Korean Society of Neonatology.2003;10(1):47-54.

PURPOSE: This report attempts to reveal the incidence and prevalence of bronchopulmonary dysplasia (BPD) and compare the severity according to preceding causes of BPD in very low birth weight (VLBW) infants. METHOD: Retrospective study was done on 293 VLBW infants who were born and admitted to neonatal intensive care unit in Samsung medical center between October, 1995 and December, 2001. Classical BPD was defined as oxygen dependency at 36 week's postmenstrual age (PMA). Ogawa BPD was defined as oxygen dependency at 28 days after birth, with respiratory distress symptoms and the change on chest X-ray finding. This classification further classified as BPD into 5 subtypes by the presence of respiratory distress syndrome (RDS), pathologic chorioamnionitis and the type of chest X-ray finding. BPD by Jobe and Bancalari was defined as oxygen dependency at 28 days after birth and classified as 3 subtypes (severe, moderate, mild) by the severity of oxygen dependency. Comparisons were made among classifications. RESULTS: Classical BPD infants were 56 (19.1%), Ogawa BPD infants were 76 (25.9 %), BPD by Jobe and Bancalari infants were 124 (42.3%). In Ogawa classification, Infants with RDS and the change on chest X-ray were 58 infants (76.4%). There was no statistical difference of mortality between each type of Ogawa BPD. In classification by Jobe and Bancalari, 35 infants (28.2%) belonged to severe BPD and 75 infants (60.5%) belonged to mild BPD. The mortality was highest in severe BPD infants but there was no statistical difference after correction by birth weight. There was no statistical correlation between Ogawa classification and classification by Jobe and Bancalari. CONCLUSION: There was no statistical difference in mortality or severity between each subtype of classifications according to the severity or preceding cause of BPD in very low birth weight infants.
Birth Weight ; Bronchopulmonary Dysplasia* ; Chorioamnionitis ; Classification ; Female ; Humans ; Incidence ; Infant* ; Infant, Newborn ; Infant, Very Low Birth Weight* ; Intensive Care, Neonatal ; Mortality ; Oxygen ; Parturition ; Pregnancy ; Prevalence ; Retrospective Studies ; Thorax

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The Relationship between the Bronchopulmonary Dysplasia and Illness Severity (SNAP).

Hyung Youl PARK ; Hyo Soo KIM ; Eun Kyung LEE ; Wook CHANG ; Sehyun KIM ; Young Jun HWANG ; Kyu Hyung LEE

Journal of the Korean Society of Neonatology.2003;10(1):39-46.

PURPOSE: The objective of this study was to evaluate differences of Scores for Neonatal Acute Physiology (SNAP) in between neonates with bronchopulmonay dysplasia (BPD) and control group, and to utilize SNAP as an early predictive tool for development of BPD. METHOD: A retrospective chart review was done for a total of 30 neonates who were admitted to Pochun CHA University neonatal intensive care unit between April, 1995 and May, 2001. A study group included 15 neonates with BPD and a control group included 15 neonates matched for gestational age and birth weight. SNAP and cumulative SNAP were obtained at 1st, 4th, 6th postnatal day for each group. A comparative analysis of cumulative SNAP scores of various parameters was done in two groups. RESULTS: SNAP of 10.86 and 6.86 were obtained at 4th postnatal day for BPD and control group, respectively (P<0.05). Cumulative SNAP for 1st, 4th, 6th postnatal day were 30.40 and 21.93 in BPD and control group (P=0.059). A comparison of cumulative SNAP of various parameters between two groups showed that respiratory rate and apnea to be significant parameters as well as for blood pressure and arterial oxygen tension in neonates with BPD. CONCLUSION: Score for Neonatal Acute Physiology could be utilized as a tool to predict the development of BPD although larger study is needed to simplify its scoring system to be used easily and better predict the development of BPD.
Apnea ; Birth Weight ; Blood Pressure ; Bronchopulmonary Dysplasia* ; Gestational Age ; Humans ; Infant, Newborn ; Intensive Care, Neonatal ; Oxygen ; Physiology ; Respiratory Rate ; Retrospective Studies

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Comparison of Treatment Outcomes in Persistent Pulmonary Hypertension of the Newborn.

Hyun Kyung PARK ; Jae Woo LIM ; Nu Lee JUN ; Ellen Ai Rhan KIM ; Ki Soo KIM ; Soo Young PI

Journal of the Korean Society of Neonatology.2003;10(1):29-38.

PURPOSE: To compare the efficacy of conventional strategy versus high-frequency oscillatory ventilation (HFOV) and inhaled nitric oxide (iNO) for the treatment of full term neonates with persistent pulmonary hypertension (PPHN). METHODS: Full term infants admitted with diagnosis of PPHN to neonatal intensive care units of Asan Medical Center from a period of 1994 to 2001 were included. The major exclusion criteria included congenital anomalies and pulmonary hypoplasia caused by various etiologies. Demographic, therapeutic responses and outcomes were compared between two study phases depending upon treatment modalities used: phase 1 (January 1994-July 1997) using conventional strategy and phase II (July 1997-December 2001) using either conventional strategy or/with iNO or HFOV with iNO. RESULTS: A total of 16 patients and 32 patients were enrolled in phase I and phase II studies, respectively. The primary diagnosis for PPHN included idiopathic (n=12), hyaline membrane disease (n=16), meconium aspiration syndrome (n=11), and sepsis (n=10). Statistically significant improvements were noted in survival, oxygenation index and alveolar- arterial oxygen tension difference during phase II than phase I study period. 75% of those infants treated with iNO showed good responses. Improvements in duration of ventilator use, oxygen supplementation, hospitalization were noted during phase II study period, however, not statistically significant compared to phase I study period. The impairments in neurodevelopmental outcomes were noted in 1 and 2 patients during phase I and phase II study periods, respectively. CONCLUSION: We conclude that HFOV and iNO are more efficacious and safe rescue mode of treatment than conventional strategy for full term infants with PPHN.
Chungcheongnam-do ; Diagnosis ; Hospitalization ; Humans ; Hyaline Membrane Disease ; Hypertension, Pulmonary* ; Infant ; Infant, Newborn* ; Intensive Care Units, Neonatal ; Meconium Aspiration Syndrome ; Nitric Oxide ; Oxygen ; Sepsis ; Ventilation ; Ventilators, Mechanical

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Analysis of Cut-off Point of Stable MicrobubbleRating Test for Predicting Neonatal Respiratory Distress Syndrome.

Min Woo KIM ; Dong Kyun PARK ; Chong Woo BAE

Journal of the Korean Society of Neonatology.2003;10(1):21-28.

PURPOSE: Early detection of RDS is imperative in treatment of neonatal respiratory distress syndrome (RDS). Shake test and stable microbubble rating (SMR) test have been commonly used for early detection of RDS. In this study, we analyzed the cut-off point of SMR test to identify whether current dianostic criteria (SMR 10/mm2) correlates with actual development of RDS. METHODS: Retrospective analysis of SMR test performed on neonates with birth weight less than 2, 500 gm or gestational age less than 37 weeks or had the symptoms of respiratory distress from 1990 to 2001. We evaluate the correlations of SMR counts and actual development of RDS and calculated the sensitivity, specificity and diagnostic accuracy at each SMR count. RESULTS: The diagnostic accuracy was considerably high when microbubble count was between 7 and 11. SMR count 9, the mean of these numbers, was determined as the cut-off point with 81.4% diagnostic accuracy. Sensitivity and specificity was 59.1% and 95.8% respectively. CONCLUSION: Current diagnostic criteria of SMR test would be effective in expecting development of RDS.
Birth Weight ; Gestational Age ; Humans ; Infant, Newborn ; Microbubbles ; Respiratory Distress Syndrome, Newborn* ; Retrospective Studies ; Sensitivity and Specificity

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