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

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

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

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

OBJECTIVE: To investigate neural correlates associated with recovery of motor function over 6 months in patients with basal ganglia (BG) stroke using acetazolamide (ACZ) stress brain-perfusion single-photon emission computed tomography (SPECT). METHODS: Medical records of 22 patients presenting first-ever BG stroke were retrospectively reviewed. Regional cerebral blood flow (CBF) and cerebrovascular reserve (CVR) were measured for 9 regions in each cerebral hemisphere (primary motor cortex, supplementary motor area, premotor cortex, prefrontal cortex, temporal lobe, parietal lobe, occipital lobe, BG, and thalamus). The Fugl-Meyer Assessment (FMA) motor score was used to assess motor function. RESULTS: After ACZ injection, CBF of all regions of interest (ROIs) increased compared with baseline. Baseline CBF of all ROIs was not significantly correlated with changes in FMA upper or lower motor score. However, multivariate analysis revealed CVR was significantly associated with change in FMA upper score in the ipsilateral primary motor cortex (R2=0.216, p=0.017), the ipsilateral parietal lobe (R2=0.135, p=0.029), and the contralateral primary motor cortex (R2=0.210, p=0.041). CONCLUSION: CVR in the bilateral primary motor cortex and ipsilateral parietal lobe was associated with restoration of upper motor function 6 months after BG stroke. SPECT is a readily available imaging modality useful in studying brain residual function in patients with BG stroke.
Acetazolamide ; Basal Ganglia* ; Brain ; Cerebrovascular Circulation ; Cerebrum ; Humans ; Medical Records ; Motor Cortex ; Multivariate Analysis ; Occipital Lobe ; Parietal Lobe ; Prefrontal Cortex ; Recovery of Function ; Retrospective Studies ; Stroke* ; Temporal Lobe ; Tomography, Emission-Computed ; Tomography, Emission-Computed, Single-Photon*

OBJECTIVE: To investigate neural correlates associated with recovery of motor function over 6 months in patients with basal ganglia (BG) stroke using acetazolamide (ACZ) stress brain-perfusion single-photon emission computed tomography (SPECT). METHODS: Medical records of 22 patients presenting first-ever BG stroke were retrospectively reviewed. Regional cerebral blood flow (CBF) and cerebrovascular reserve (CVR) were measured for 9 regions in each cerebral hemisphere (primary motor cortex, supplementary motor area, premotor cortex, prefrontal cortex, temporal lobe, parietal lobe, occipital lobe, BG, and thalamus). The Fugl-Meyer Assessment (FMA) motor score was used to assess motor function. RESULTS: After ACZ injection, CBF of all regions of interest (ROIs) increased compared with baseline. Baseline CBF of all ROIs was not significantly correlated with changes in FMA upper or lower motor score. However, multivariate analysis revealed CVR was significantly associated with change in FMA upper score in the ipsilateral primary motor cortex (R2=0.216, p=0.017), the ipsilateral parietal lobe (R2=0.135, p=0.029), and the contralateral primary motor cortex (R2=0.210, p=0.041). CONCLUSION: CVR in the bilateral primary motor cortex and ipsilateral parietal lobe was associated with restoration of upper motor function 6 months after BG stroke. SPECT is a readily available imaging modality useful in studying brain residual function in patients with BG stroke.
Acetazolamide ; Basal Ganglia* ; Brain ; Cerebrovascular Circulation ; Cerebrum ; Humans ; Medical Records ; Motor Cortex ; Multivariate Analysis ; Occipital Lobe ; Parietal Lobe ; Prefrontal Cortex ; Recovery of Function ; Retrospective Studies ; Stroke* ; Temporal Lobe ; Tomography, Emission-Computed ; Tomography, Emission-Computed, Single-Photon*

PURPOSE: To evaluate two different heel lancet device in terms of pain response and success of the procedure in the preterm infants undergoing heel puncture. METHODS: 100 preterm infants undergoing capillary blood gas analysis or capillary bilirubin monitoring underwent heel puncture, were randomly allocated to blood sampling from the heel with either a conventional manual lancet or an automatic incision device. Primary outcome measures included the Premature Infants Pain Profile (PIPP) score, total duration of procedure, number of heel puncture and number of bruise. The pain response was evaluated using PIPP score and the effectiveness was evaluated using three criteria: total duration of blood sampling, number of puncture, bruising of the heel or ankle. Statistical analysis was performed using the SPSS ver. 13.0 program. Difference between the groups were analysed with t test (continuous variables) and the Chi square test or Fisher test (categorical variables). RESULTS: The mean PIPP score was 4.91 for the automatic lancet group compared with 5.84 for the conventional manual lancet group (P=0.0255).The number of pain scores above 7 during blood collection did not differ between two groups (P=0.2167). The procedure took less time to perform in the automatic lancet group (mean, 30.69 seconds) than in the conventional lancet group (mean, 48.92 seconds) (P<0.0001). CONCLUSION: This study demonstrated that the automatic lancet device causes less pain and a shorter procedure time than the conventional manual lancet in preterm infants undergoing heel puncture. On the basis of these results the automatic lancet device is very useful method for blood collection in preterm infants by heel puncture.
Animals ; Ankle ; Bilirubin ; Blood Gas Analysis ; Capillaries ; Contusions ; Heel ; Humans ; Infant, Newborn ; Infant, Premature ; Outcome Assessment (Health Care) ; Punctures

PURPOSE: To evaluate two different heel lancet device in terms of pain response and success of the procedure in the preterm infants undergoing heel puncture. METHODS: 100 preterm infants undergoing capillary blood gas analysis or capillary bilirubin monitoring underwent heel puncture, were randomly allocated to blood sampling from the heel with either a conventional manual lancet or an automatic incision device. Primary outcome measures included the Premature Infants Pain Profile (PIPP) score, total duration of procedure, number of heel puncture and number of bruise. The pain response was evaluated using PIPP score and the effectiveness was evaluated using three criteria: total duration of blood sampling, number of puncture, bruising of the heel or ankle. Statistical analysis was performed using the SPSS ver. 13.0 program. Difference between the groups were analysed with t test (continuous variables) and the Chi square test or Fisher test (categorical variables). RESULTS: The mean PIPP score was 4.91 for the automatic lancet group compared with 5.84 for the conventional manual lancet group (P=0.0255).The number of pain scores above 7 during blood collection did not differ between two groups (P=0.2167). The procedure took less time to perform in the automatic lancet group (mean, 30.69 seconds) than in the conventional lancet group (mean, 48.92 seconds) (P<0.0001). CONCLUSION: This study demonstrated that the automatic lancet device causes less pain and a shorter procedure time than the conventional manual lancet in preterm infants undergoing heel puncture. On the basis of these results the automatic lancet device is very useful method for blood collection in preterm infants by heel puncture.
Animals ; Ankle ; Bilirubin ; Blood Gas Analysis ; Capillaries ; Contusions ; Heel ; Humans ; Infant, Newborn ; Infant, Premature ; Outcome Assessment (Health Care) ; Punctures