A case of spontaneous resolution of nonimmune hydrops fetalis

Indoor average 222Rn concentrations have been measured in more than 400 houses in Istanbul city. The average radon concentrations in the houses were found to be between 10 Bq m-3 and 260 Bq m-3 and the mean value was 50 Bq m-3.     

Ectopic neuroglial tissue associated with intrapulmonary congenital cystic adenomatoid malformation

BACKGROUND: Few cases of ectopic neurological tissue have been reported in the lung. The aim of the present study was to give a brief overview of these cases and to examine an additional case of intrapulmonary neuroglial heterotopia. We have identified only sixteen similar cases in the literature. CASE DESCRIPTION: The object of our study was a male fetus of Asian parents at the 23rd week of gestation, in which ultrasound tests revealed the presence of anterior encephalocele. Routine postmortem examination of lung samples showed neuroglial tissue and a congenital adenomatoid cystic malformation of type II. The lesion was made up of multiple small cysts lined with columnar or ciliated cuboidal epithelium. A possible link between adenomatoid malformation and intrapulmonary neurological tissue has not so far been reported in the literature. Immunohistochemical analysis showed the presence in the pulmonary parenchyma of neuronal cells (neuron-specific enolase positive), astrocytes (glial fibrillary acidic protein positive) and intra-alveolar squamous cells (citokeratines positive), indicative of fetal aspiration of amniotic fluid. CONCLUSIONS: There are several possible explanations for the presence of intrapulmonary neuroglial heterotopia: fetal aspiration, neural crest migration defects or vascular embolization with implantation. However, in the view of the microscopic findings and at the same time recognizing the intrapulmonary aspiration of amniotic fluid, the authors maintain that the most likely explanation for the heterotopia is that of consequential multiple malformations. Moreover neuroglial ectopy and cystic adenomatoid congenital malformation of the lung could have appeared simultaneously, due to embryologic insult between the 4th and the 20th week of gestation.     

Bilateral and multilobar cystic adenomatoid malformation of the lung

In this study we sought to evaluate the potential of rat tail replantation as a tool for very-small-vessel microvascular anastomoses. We used 10 adult Sprague-Dawley rats. The tail was completely amputated 2.0-cm distal to the base of the tail. Then the tail was replanted with anastomoses of two superficial dorsal veins from both sides and one artery. All 10 replanted tails were pink, viable, and normal-appearing at all daily inspections performed from the first to the fourteenth postoperative days. This model can provide a training tool for the acquisition of superior microvascular surgical technique for the repair of very small vessels that stimulate digital replantation.     

Case report--a neonate with nonimmune hydrops fetalis

AIM: To investigate the waste of laboratory reagents which resulted from the process of ordering biochemistry profiles. METHODS: The frequency of measurement of 15 analytes was recorded during the six months before the introduction of a system of discretionary requesting and analysis of samples (high capacity multichannel discrete analyser), and also during the same six month period one year and two years after its introduction. RESULTS: The frequency of measurement of 10 of the 15 analytes decreased during the six month period one year after the change to discretionary testing. The remaining five analytes were measured up to 22% more frequently. There was an overall decrease in the measurement of biochemical tests by 31,359. This created an annual cost saving of 7124 pounds. In the second year five analytes still continued to be measured less frequently than originally but the remaining 10 analytes were measured more frequently. This resulted in an overall increase in the measurement of biochemical tests by 53,678 compared with the six month period before discretionary analysis. The pattern of requests was similar during both periods of discretionary requesting studied and as a result, a small annual cost saving of 1672 pounds was again made. CONCLUSION: Discretionary requesting and analysis of tests may eliminate the measurement of clinically unnecessary test which had previously resulted from the processes of ordering tests. These cost savings may be rapidly eroded by an increase in the laboratory workload.     

Successful outcome with serial amniocenteses for polyhydramnios complicating cystic adenomatoid malformation of the lung

Hydrops fetalis secondary to congenital cystic adenomatoid malformation of the fetal lung is almost invariably a lethal condition. This poor prognosis may be attributed to the combined effects of tissue compression from the thoracic space occupying lesion and premature delivery of a hydropic baby following rupture of the membranes in the presence of polyhydramnios. We describe a successful outcome with serial amniocenteses. Partial in utero resolution of the lung lesion with complete return to normal of the aberrant liquor volume was documented, and a live infant was delivered at term.     

Antenatal diagnosis and surgical management of congenital cystic adenomatoid malformation of the lung

We experienced 12 cases of congenital cystic adenomatoid malformation of the lung (CCAM) including 6 cases diagnosed antenatally. They were classified into three groups according to the clinical manifestations. Group A was associated with hydrops fetalis (n = 3), group B presented with respiratory distress symptoms after birth (n = 6), and group C showed no respiratory symptoms (n = 3). All cases of group A were lost because of hydrops and respiratory failure due to pulmonary hypoplasia. Because a compression of the mass is thought to be a cause of hydrops, this group is considered to be a good indication for fetal treatment. All cases of group B showed progressive respiratory symptoms a few days after birth which were successfully treated surgically. In 2 of 3 cases of group C, the lesions decreased in size both antenatally and postnatally. We conclude that serial sonographic evaluations for fetal CCAM are important. If the fetus develops hydrops, fetal surgery is to be considered. If not, however, fetal surgery should not be done, because some lesions can shrink in size, or even disappear, while others can be treated successfully after birth by lobectomy or even segmentectomy.     

Marked twin-twin transfusion in the absence of nonimmune hydrops

The precise pathophysiology of development of nonimmune hydrops in either the recipient or donor twin associated with twin-twin transfusion syndrome is not entirely clear. At times the recipient twin may develop nonimmune hydrops, but at other times the donor twin, and infrequently both, may develop this ominous complication. We present an unusual cases of this syndrome in which discordant twins, despite neonatal hematocrit levels of 86 and 21% were both nonhydropic and discuss the possible underlying pathophysiology of this occurrence.     

Extralobar pulmonary sequestration mimicking cystic adenomatoid malformation in prenatal sonographic appearance and histological findings

An infant girl with extralobar pulmonary sequestration (PS) composed of congenital cystic adenomatoid malformation (CCAM)-like structure is presented. Initially, the antenatal sonographic findings indicated CCAM. The macroscopic findings of the resected specimen were compatible with extralobar PS; however, the microscopic findings showed cystic structure mimicking type II CCAM. The combination of PS and CCAM is rare, and it is likely that the embryological origin is common to both. There is confusion in the classification of these two congenital anomalies. In this report, the histological and sonographic findings of PS and CCAM are discussed.     

Pleural pneumatocoeles mimicking congenital cystic adenomatoid malformation of the lung. A case report

The beta-specific mRNA were identified in the suppressor cell-line of murine and suppressor hybridoma of mouse origin by mRNA-DNA hybridization and isolation of cDNA with DNA and antibody probes.     

A series of 17 cases of congenital cystic adenomatoid malformation of the lung: management and outcome

During a 5-year-period, 17 cases of congenital adenomatoid malformation of the lung were observed in the Department of Pediatric Surgery of Bologna University-S. Orsola-Malpighi Hospital; 16 cases were diagnosed in utero by ultra-sound. 13 patients had prenatal diagnosis between 21 and 23 weeks of gestation; 2 cases were diagnosed at 25 weeks and another one at 26 weeks. The other case came from another hospital, and the diagnosis was accidentally detected postnatally. In 3 cases the parents decided to terminate the pregnancy 3 cases had spontaneous improvement during the last weeks of gestation, and were perfectly normal at birth. One case was lost to follow-up; all remaining patients underwent resection. Among these patients only one was a nonsurvivor, while 9 have completely recovered. However, one patient developed persistent pulmonary hypertension, which could successfully be treated with drugs, another underwent laser treatment for laryngeal stenosis due to intubation. A special epidemiological information: 13 out 17 cases (76.5%) were female.     

Spontaneous disappearance of a renal arteriovenous malformation

We describe herein a case of complete spontaneous disappearance of a congenital arteriovenous malformation (AVM). A 28-year-old male was hospitalized for right flank pain and gross hematuria, followed by bladder tamponade. To improve the patient's symptoms, bladder irrigation was performed. Cystoscopy demonstrated bloody urine from the right ureteral orifice. Right selective renal arteriography demonstrated tortuous, coiled vascular channels with early filling of the renal vein. Thus, right renal AVM was diagnosed. However, the patient refused further treatment and was discharged. One year later, massive hematuria recurred with bladder tamponade and the patient was rehospitalized. Renal arteriography did not show any evidence of AVM and there has been no hematuria since.     

Spontaneous intracranial hypotension: headache with a reversible Arnold-Chiari malformation

Spontaneous intracranial hypotension is characterized by severe postural headache in the setting of low CSF pressure, usually attributed to a cryptic CSF leak. We report a patient whose prolonged refractory headache was characterized by the clinical symptoms of occipital neuralgia, but was also associated with the radiographic appearance of an Arnold-Chiari malformation, type I and low CSF pressure. After extensive diagnostic evaluation, CT cisternomyelography ultimately demonstrated a CSF leak at the C2 vertebral level. Symptomatic relief was sustained only with long-term theophylline administration. The apparent Arnold-Chiari malformation resolved with treatment of the low CSF pressure.     

Analysis of cardiotocographic recordings preceding intrauterine death of fetus with non-immunological hydrops

Continuous, 12 hours-long cardiotocographic recordings preceding intrauterine death of fetus (in 29 week) with non immunological hydrops were analysed. The features of CTG recordings were: absence of short and long-term variability, absence of tachycardia, presence of late decelerations, and terminal bradycardia of short duration.     

The use of "nonanatomic" pulmonary resection in infants with extensive congenital adenomatoid malformation of the lung

In three infants with extensive congenital adenomatoid malformation of the lung, a method of nonanatomic pulmonary resection permitted preservation of small superiorly positioned pulmonary remnants. These remnants then demonstrated a surprising capacity for expansion, growth, and function during follow-up periods of 5 years, 9 months; 7 years, 6 months; and 19 years. Thus far, this small experience indicates that this method of management does not appear to entail greater short- or long-term risks than the alternative procedure of pneumonectomy.     

Spontaneous resolution of cholelithiasis in an infant

Cholelithiasis is an extremely unusual finding in infancy. Hemolytic disease, bile duct abnormalities, intestinal malabsorption have been implicated. Recently, an association of prematurity, bronchopulmonary dysplasia, total parenteral nutrition and furosemide has been reported. Rarely, sonographic discovery of idiopathic gallstones and their subsequent spontaneous disappearance has been reported. We report a case of an 11-week-old infant whose initial sonogram displayed evidence of true gallstone, that later showed resolution on follow-up examinations. In the absence of clinical or imaging evidence of biliary tract disease, we suggest an initial conservative management and follow-up sonograms.     

Spontaneous rupture of benign cystic teratomas

Spontaneous preoperative rupture and/or perforation of an adjacent organ by a benign cystic teratoma is rare. Between 1952 and 1979 there were 4 documented cases at The Johns Hopkins Hospital. The incidence, etiology, and pathology of this complication are discussed.