This is a case report of a cystic hygroma with polycystic kidney in a fetus which was suspected by ultrasonography and was confirmed by autopsy. Recently, we have experienced this case in 25-year old woman repeatedly and we report that with a brief review of relevant literature.
Adult ; Autopsy ; Female ; Fetus ; Humans ; Lymphangioma, Cystic* ; Polycystic Kidney Diseases* ; Ultrasonography

Cutaneous lung tissue heterotopia is a very rare disorder where mature lung tissues develop in the skin. This is only the second known report of cutaneous lung tissue heterotopia, with the first by Singer et al. in 1998. A newborn infant had a hemangioma-like, freely movable mass connected to the anterior aspect of the sternal manubrium. Pathologic findings showed mature lung tissues with bronchi, bronchioles, and alveoli through the dermis and subcutis, and it was diagnosed as cutaneous lung tissue heterotopia. Cutaneous lung tissue heterotopia is hypervascular, so grossly it looks like a hemangioma. It can be differentiated from pulmonary sequestration, teratoma, bronchogenic cyst, and branchial cleft cyst by histology and the location of the mass. We describe the clinical, radiologic, and pathologic findings of a cutaneous lung tissue heterotopia, the first reported in Korea.
Branchioma/pathology/surgery ; Bronchogenic Cyst/pathology/surgery ; Choristoma/*pathology/surgery ; Humans ; Infant, Newborn ; *Lung ; Magnetic Resonance Imaging ; Male ; Republic of Korea ; Skin Diseases/*pathology/surgery ; Skin Neoplasms/pathology ; Tomography, X-Ray Computed

PURPOSE: In this study, the risk factors of failure of ibuprofen treatment in preterm infants with hemodynamically significant patent ductus arteriosus (hsPDA) were investigated. METHODS: Among 403 preterm infants (<32 weeks gestation) born between January 2010 and December 2012, 125 infants treated with ibuprofen for hsPDA were retrospectively reviewed. The preterm infants were divided into the following groups according to their response to the 1st and 2nd cycles of ibuprofen treatment: responder groups I and II, closure of the ductus arteriosus after the 1st and 2nd cycles of ibuprofen treatment; and non-responder groups I and II, persistency of hsPDA after the 1st and 2nd cycles of ibuprofen treatment. RESULTS: One hundred twenty five infants were enrolled in the study: 74 in responder group I, 51 in non-responder group I, 14 in responder group II, and 22 in non-responder group II. In non-responder group I, the gestational age and birth weight were smaller, the postnatal steroid treatment was more frequent, and the duration of mechanical ventilation and the days spent in the hospital were prolonged.I n non-responder group II, the gestational age and birth weight were smaller, the diameters of the ductus arteriosus were larger, and the inotropics use was more frequent. CONCLUSION: Failure of ibuprofen treatment of hsPDA is associated with the diameter of the ductus arteriosus and with inotropics use. Obtaining data regarding these is expected to help in determining if early direct surgical ligation is needed.
Birth Weight ; Ductus Arteriosus ; Ductus Arteriosus, Patent* ; Gestational Age ; Humans ; Ibuprofen* ; Infant ; Infant, Newborn ; Infant, Premature* ; Ligation ; Respiration, Artificial ; Retrospective Studies ; Risk Factors*

PURPOSE: Thyroid dysfunction is common in preterm infants. Congenital hypothyroidism causes neurodevelopmental impairment, which is preventable if properly treated. This study was conducted to describe the characteristics of thyroid dysfunction in very low birth weight infants (VLBWIs), evaluate risk factors of hypothyroidism, and suggest the reassessment of thyroid function with an initially normal thyroid-stimulating hormone (TSH) as part of a newborn screening test. METHODS: VLBWIs (January 2010 to December 2012) were divided into two groups according to dysfunction-specific thyroid hormone replacement therapy, and associated factors were evaluated. RESULTS: Of VLBWIs, 246 survivors were enrolled. Only 12.2% (30/246) of enrolled subjects exhibited thyroid dysfunction requiring thyroid hormone replacement. Moreover, only one out of 30 subjects who required thyroid hormone treatment had abnormal thyroid function in the newborn screening test with measured TSH. Most of the subjects in the treatment group (22/30) exhibited delayed TSH elevation. Gestational age, Apgar score, antenatal steroids therapy, respiratory distress syndrome, patent ductus arteriosus, sepsis, intraventricular hemorrhage, postnatal steroids therapy, and duration of mechanical ventilation did not differ between the two groups. Birth weight was smaller and infants with small for gestational age were more frequent in the treatment group. CONCLUSION: Physicians should not rule out suggested hypothyroidism, even when thyroid function of a newborn screening test is normal. We suggest retesting TSH and free thyroxine in high risk preterm infants with an initially normal TSH level using a newborn screening test.
Apgar Score ; Birth Weight ; Congenital Hypothyroidism ; Ductus Arteriosus, Patent ; Gestational Age ; Hemorrhage ; Hormone Replacement Therapy ; Humans ; Hypothyroidism ; Infant ; Infant, Newborn ; Infant, Premature* ; Infant, Very Low Birth Weight* ; Mass Screening ; Neonatal Screening ; Respiration, Artificial ; Respiratory Therapy ; Risk Factors ; Sepsis ; Steroids ; Survivors ; Thyroid Function Tests ; Thyroid Gland* ; Thyrotropin ; Thyroxine

Diffuse neonatal hemangiomatosis (DNH) is characterized by multiple capillary or cavernous hemangiomas on the skin and internal organs occurring during the neonatal period. It is a life-threatening condition due to high-output heart failure with a mortality rate of 60-85% without proper treatment. The areas that are most commonly involved include the skin (100%), liver (64-100%), and central nervous system (52%). Corticosteroids are the drugs of choice as an initial treatment and have a response rate of 30-60%. We present here a case of a newborn baby with multiple hemangiomas on her skin (scalp, lips, neck, back, shoulder, arm, buttock, and leg), brain (right cerebellum, pons, and medulla oblongata), lungs, liver, kidney, and bones. She suffered from 6th, 7th, 9th, 10th, and 12th cranial nerve palsy resulting from hemorrhage of the hemangiomas in the brain. The first-line treatment of prednisolone (4 mg/kg/day) was not effective and propranolol (2 mg/kg/day) was administered as a second-line treatment. After 2 weeks of treatment, the hemangiomas had decreased in size with no associated acute hemorrhage. The infant is now 10 months old and both the multiple hemangiomas and cranial nerve palsy have improved. Propranolol was effective without significant adverse effects in treating DNH resistant to corticosteroids.
Abducens Nerve Diseases ; Adrenal Cortex Hormones ; Arm ; Brain ; Buttocks ; Capillaries ; Central Nervous System ; Cerebellum ; Cranial Nerve Diseases* ; Cranial Nerves* ; Facial Paralysis ; Heart Failure ; Hemangioma ; Hemangioma, Cavernous ; Hemorrhage ; Humans ; Infant ; Infant, Newborn ; Kidney ; Lip ; Liver ; Lung ; Mortality ; Neck ; Pons ; Prednisolone ; Propranolol* ; Shoulder ; Skin

Our purpose was to compare the safety and efficacy of intravaginal misoprostol versus dinoprostone vaginal tablet for induction of labor at term pregnancies. Two hundred three patients with indications for induction of labor at term were randomly assigned to receive either intravaginal misoprostol or dinoprostone vaginal tablet. Fifty micrograms of misoprostol was placed in the posterior vaginal fornix every six hours, with a potential maximum of four doses. 3 milligrams of dinoprostone vaginal tablet was placed in the posterior vaginal fornix every eight hours, with a potential maximum of three doses. Further medication was withheld with the occurrence of spontaneous rupture of membrane, entry into adequate contraction pattern(twenty second sustained with two or more frequent uterine contraction in 10 minutes), nonreassuring FHR tracing, or delivery. Artificial membrane rupture with both study protocol was done at the discretion of the attending physician. After membrane rupture, in the cases of failure of active labor or arrest of dilation, oxytocin was administerated. Among those evaluated, 100 received misoprostol and 102 received dinoprostone. The average interval from start of induction to vaginal delivery was shorter in the misoprostol group(784.7 +/- 389.3 min) than in the dinoprostone group(988.3 +/- 369.5 min)(p<0.01). There was no significant difference in change of Bishop score between the two groups. No statistically significant differences were noted between two groups in case of need for oxytocin and oxytocin total dose, but significant difference was noted between two groups in case of indication for oxytocin augmentation. There were no significant differences in the routes of delivery. Intravaginal administration of misoprostol appears to be as effective as dinoprostone vaginal tablet for labor induction at full term pregnancies. Complications associated with prostaglndin administration were not statistically different between the two treatment groups.
Administration, Intravaginal ; Dinoprostone* ; Humans ; Membranes ; Membranes, Artificial ; Misoprostol* ; Oxytocin ; Pregnancy* ; Rupture ; Rupture, Spontaneous ; Uterine Contraction ; Vaginal Creams, Foams, and Jellies*

PURPOSE: This study investigated the effects of early enteral feeding on the morbidities of extremely low birth weight infants (ELBWI) weighing less than 1,000 g. METHODS: We conducted a retrospective review of the medical records of sixty one ELBWI who were admitted to the neonatal intensive care unit of Inje University Busan Paik Hospital from January 2007 to October 2009. ELBWI were divided into two groups; the control group included ELBWI from January 2007 to March 2008, for whom enteral feeding was started beyond 3 days and the early feeding group included ELBWI from April 2008 to October 2009, for whom enteral feeding was started within 3 days. RESULTS: Gestational age and birth weight did not differ between the two groups. In the early feeding group, start day of enteral feeding (control group vs. early feeding group; 7+/-2days vs. 2+/-1days), time to achieve full enteral feeding (68+/-6 days vs. 22+/-2 days), and the duration of parenteral nutrition (58+/-6 days vs. 22+/-2 days) were significantly shorter, and weight gain at postnatal day 28 was significantly higher than that of the control group (P<0.001). No differences were observed in the incidence of sepsis and necrotizing enterocolitis and duration of hospitalization; however, the incidence of total parenteral nutrition induced cholestasis (44% vs. 7%) and bronchopulmonary dysplsia (78% vs. 24%) was significantly lower in the early feeding group. CONCLUSION: Early enteral feeding in ELBWI shortened the time to achieve full enteral feeding, improved weight gain, and decreased the incidence of brochopulmonay dysplasia and cholestasis.
Birth Weight ; Cholestasis ; Enteral Nutrition ; Enterocolitis, Necrotizing ; Gestational Age ; Humans ; Incidence ; Infant ; Infant, Low Birth Weight ; Infant, Newborn ; Intensive Care, Neonatal ; Medical Records ; Parenteral Nutrition ; Parenteral Nutrition, Total ; Retrospective Studies ; Sepsis ; Weight Gain

BACKGROUND AND OBJECTIVES: We report our initial experience with percutaneous transvenous closure of atrial septal defects (ASD). MATERIALS AND METHOD: Between September 1997 and May 2000, we attempted transcatheter closure of ASD in 18 patients using CardioSEALTM (8), STARFlexTM (4) and Amplatzer septal occluder (6). The ages of patients ranged from 4.5-64.8 (mean 32.8) years, body weight ranging from 16-76 (mean 51) kg, Qp/Qs ratio from 1.3-3.4 (mean 2.2). RESULTS: Embolization of device occurred in two patients; right pulmonary artery in one and left atrium in the other. In one patient, the device slipped into the right atrium before detachment. After retrieval of the device, the defect seemed too large for transcatheter closure. There were no other complications apart from a transient aggravation of pre-existing atrial premature beats in two patients. There was no significant size difference between the data measured by transthoracic and transesophageal echocardiography. The stretched ASD diameter was larger (5.1 3.2 mm) than the size measured by transesophageal echocardiography. In the remaining 15 patients, complete closure of defects was confirmed by transthoracic echocardiography on the 1 day or 1 month follow-up. During the same period, transcatheter closure of patent foramen ovale(PFO) was also attempted in 7 patients with stroke. The guidewire could not be passed in 2 of the patients. In the other 5 patients, transcatheter closure was successfully performed without any problems. Though the follow-up period may have been short, no patients were found with further stroke attack. CONCLUSION: Transcatheter closure of ASD can be performed with high efficiency and safety if patient selection is adequate. The indication for ASD closure can be extended to patients with larger defects. Transcatheter closure of PFO is an easy and safe procedure, but the indications of PFO closure in patients with stroke is still unclear. Further evaluation is necessary for long-term results.
Body Weight ; Cardiac Complexes, Premature ; Echocardiography ; Echocardiography, Transesophageal ; Follow-Up Studies ; Heart Atria ; Heart Septal Defects, Atrial* ; Humans ; Patient Selection ; Pulmonary Artery ; Septal Occluder Device ; Stroke

Congenital epulis is a rare benign tumor occurring on the anterior maxillary gingiva, also known as granular cell tumor of the newborn or Neumann's tumor, which is seen only in the newborn and is different from other granular cell tumors. Congenital epulis occurs exclusively in female newborns eight to ten fold higher than in males. It can protrude out of the newborn's mouth to prevent normal closure of mouth and interfere with respiration or feeding. The treatment of choice for large symptomatic epulis is simple surgical resection. Wide surgical excision is not required, because no recurrences have been reported. This report describes a case of congenital epulis occurring on the mandibular gingiva, and typical immunohistochemical stain findings.
Female ; Gingiva ; Gingival Diseases ; Gingival Neoplasms ; Granular Cell Tumor ; Humans ; Infant, Newborn ; Male ; Mouth ; Recurrence ; Respiration

PURPOSE: The purpose of this study was to identify factors influencing premature infants who are small for their gestational ago. METHODS: The medical records of 1,010 premature infants of 26 to 35 weeks of gestational age born at Il-Sin Christian Hospital, Busan from January 2000 to August 2006 were reviewed. We collected data on gestational age, birth weight, infant gender, birth order, maternal age and previous abortion history and analyzed the factors influencing premature infants who were small for their gestational ago at birth. RESULTS: In our study more female than male (P=0.042) in premature infants who were small for their gestational ago were born from mothers aged younger than 20 or older than 35 (P=0.041). But association between smallness for gestational age and birth order or maternal previous abortion history was statistically insignificant (P=0.228, P=0.129). CONCLUSION: Considering the association of birth weight and the survival rate of premature infants, it is thought that maternal age had an influence on the survival rate of premature infants. Social and political support to lower the teenager pregnancy and older pregnancy is expected to increase the survival rate of premature infants and the birth of healthy normal neonates.
Abortion, Induced ; Adolescent ; Birth Order ; Birth Weight* ; Busan ; Female ; Gestational Age ; Humans ; Infant ; Infant, Newborn ; Infant, Premature* ; Male ; Maternal Age ; Medical Records ; Mothers ; Parturition* ; Pregnancy ; Survival Rate