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PubMed1999

Do the structures of big ET-1 and big ET-3 adopt a similar overall fold? Consequences for endothelin converting enzyme specificity.

Big ET-1 and big ET-3 are precursor peptides which render endothelin-1 (ET-1) and endothelin-3 (ET-3) relatively unreactive and resistant to proteolytic cleavage. Big ET-1 is cleaved in vivo by ECE-1 (endothelin-converting enzyme), and big ET-3 is also cleaved but apparently to a significantly lesser extent by this enzyme. To shed light on the relation between structure and function, circular dichroism (CD) spectroscopy and homology modeling were used to determine whether big ET-1 and big ET-3 adopt similar secondary and tertiary structures. Analyses of the CD spectra and thermal denaturation indicate they have similar secondary structures and thermal stabilities. Superposition of the modeled coordinates of both peptides indicates that they can adopt the same overall fold except in the C-terminal residues, 34-38 in big ET-1 and 34-41 in big ET-3. This region corresponds to an area of complete sequence heterogeneity between the two peptides. A model has been developed which has a loop for residues 27-30 (HVVP in big ET-1), which have previously been demonstrated to be essential for eliciting efficient hydrolysis of the W21-V22 bond in big ET-1 and which have the sequence QTVP in big ET-3. Differences in affinity between big ET-1 and big ET-3 for ECE-1 thus appear to be due solely to sequence variations in the local region of the cleavage site.

Amino Acid Sequence↗
PubMed1992

Cloning of a big tau microtubule-associated protein characteristic of the peripheral nervous system.

Microtubule-associated protein tau consists in brain of a series of isoforms of 48- to 67-kDa apparent molecular mass that are encoded by mRNAs of approximately 6 kilobases (kb) and that are generated from a single gene by alternative splicing. Previously, a tau-like protein of 110-kDa apparent molecular mass was described in peripheral ganglia and in peripheral neuronlike cell lines. We now report the cloning and sequencing of a rat cDNA encoding this big tau. The corresponding protein contains sequence identical to the longest of the previously cloned small tau isoforms but with an additional 254 amino acid insert in the amino-terminal half. Big tau is produced from an 8-kb mRNA generated by alternative splicing from the same gene that encodes small tau. Production of big tau from the cloned sequence gives a protein of 110-kDa apparent molecular mass that aligns on SDS/PAGE with big tau protein extracted from peripheral ganglia. RNA blots show that in peripheral ganglia from adult rats only the 8-kb mRNA band corresponding to big tau is found, whereas in ganglia from newborn rats both 6- and 8-kb tau mRNA bands are found. In tissues from the central nervous system only the 6-kb mRNA band can be detected. Big tau protein is therefore produced specifically in the peripheral nervous system, and it will be interesting to see whether further molecular differences between the two major divisions of the vertebrate nervous system will be discovered.

Amino Acid Sequence↗
PubMed2002

[Re-cloning of THP gene and construction of high efficient expression yector of Volvariella volvacea].

PCR technique was used for amplifying THP gene in an unknown vector with primer AFP1 and AFP2. Then THP gene was ligated to pGEM T-Vector to be the plasmid pGTHP4. The plasmid pCAMBIA1301 was digested with restriction enzyme BstE II and Nco I, and digestion product was separated with 1% of agarose gel, then big fragment containing promoter was isolated and purified with the Agarose Gel DNA Extraction Kit. At the same way, the plasmid pGTHP4 was digested with restriction enzyme BstZ I and Nco I, and the small fragment containing THP gene was purified from 1% agarose gel with the Agarose Gel DNA Extraction Kit. The big fragment and the small fragment were ligated at Nco I digested cohesive-end. The ligation product was re-ligated to be cyclic plasmid by addition to a specific adapter, resulting in the pCTH823, a expression vectorof V. volvacea.

Agaricales↗
PubMed2002

[Acute symptomatic hyponatremia in a 70-year-old patient. Insufficient ADH secretion syndrome].

CASE REPORT: A 70-year-old male was admitted to the hospital with anorexia, nausea, headache, and apathy. Serologically remarkable were a severe hyponatremia, serum hypoosmolality and urine hyperosmolality with normal renal function. After excluding adrenal insufficiency, hypothyroidism and other possible causes of hypoosmolar hyponatremia, the diagnosis of a syndrome of inadequate ADH secretion (SIADH) was confirmed through a nonsuppressed ADH level and a rise in of serum sodium after fluid restriction. Magnetic resonance imaging showed a small, chronic, traumatic subdural hematoma which was responsible for the SIADH in our patient. CONCLUSION: This etiologic correlation was reported in literature by many authors. However, these SIADHs were associated with a big subdural hematoma which needed neurosurgical intervention in most of the cases. Our patient is unique in that his symptoms were caused by a small subdural hematoma, which did not need surgical intervention. The patient was treated with cautious saline infusion, administration of a loop diuretic and fluid restriction with improvement of both his clinical and metabolic state. As large water loads may result in severe hyponatremia in these patients, moderate fluid restriction appears to be effective in long-term management even after stabilization/normalization of serum sodium. There are no sufficient data about the incidence of asymptomatic hyponatremia due to SIADH resulting from minor head injury, because most of these patients are asymptomatic and therefore they do not receive clinical attention.

Aged↗
PubMed2007

Deficiency of the Cockayne syndrome B (CSB) gene aggravates the genomic instability caused by endogenous oxidative DNA base damage in mice.

The Cockayne syndrome B protein (CSB) has long been known to be involved in the repair of DNA modifications that block the RNA polymerase in transcribed DNA sequences (transcription-coupled repair). Recent evidence suggests that it also has a more general role in the repair of oxidative DNA base modifications such as 7,8-dihydro-8-oxo-2'-deoxyguanosine (8-oxoG). In mammalian cells, 8-oxoG is a substrate of the repair glycosylase OGG1. Mice without this enzyme accumulate 8-oxoG in the genome and have elevated spontaneous mutation rates. To elucidate the role of CSB in the prevention of mutations by oxidative DNA base damage, we have generated mice that are deficient in Csb or Ogg1 or both genes and carry a non-transcribed bacterial lacI gene for mutation analysis (Big Blue mice). Our results indicate that the overall spontaneous mutation frequencies in the livers of Csb(m/m)/Ogg1-/- -mice are elevated not only compared with heterozygous control mice (factor 3.3), but also with Ogg1-/- -animals (factor 1.6). Sequence analysis revealed that the additional mutations caused by CSB deficiency in an Ogg1-/- background are mostly G:C to T:A transversions and small deletions. For all mouse strains, the background levels of oxidative purine modifications in the livers correlate linearly with the numbers of G:C to T:A transversions observed. The data indicate that CSB is involved in the inhibition of mutations caused by spontaneous oxidative DNA base damage in a non-transcribed gene.

Animals↗
PubMed2002

Diabetes care in Kilimanjaro region: clinical presentation and problems of patients of the diabetes clinic at the regional referral hospital-an inventory before structured intervention.

BACKGROUND: In order to improve care for diabetic patients in a developing country at the regional referral hospital for the Northern zone of Tanzania, a specialized clinic was established in November 1996. AIM: The aim of this study was to provide a situational analysis about the problems of the diabetic patient population treated at the hospital. For all patients who registered at the clinic a questionnaire was completed about personal data, diabetes history, symptoms, treatment, eating habits and psycho-social aspects of the disease. Results from patients who registered between November 1996 and December 1998 were analysed. RESULTS: Data from 474 diabetic patients (46% female) were obtained. Mean age was 53.75 (+/- 16; range 4-88 years), 15% were classified Type 1, 75% Type 2 and 10% remained unclassified. A body mass index of < 25 was recorded in > 80% of the Type 1 and approx. 50% of the Type 2 patients. Among complications, numbness of the legs was mentioned by 44%, hypertension was diagnosed in about 25%, retinopathy in 14%, foot ulcers in 10% and nephropathy in 7.5% of the patients. About 50% of the patients saw their disease as a big physical and psychological problem. Monthly cost for an average insulin-treated patient equalled around 25% of the minimal wage. CONCLUSION: Diabetes care in a developing country needs to address the specific background of the patient population, their needs, the medical problems and the social constraints. Active participation of the patients can help to overcome some of the difficulties.

Adolescent↗
PubMed2002

Antiprolactin autoantibodies are associated with hyperprolactinemic status in men infected with human immunodeficiency virus.

High serum prolactin (PRL) levels and even hyperprolactinemia are a common finding in human immunodeficiency virus (HIV) infection. However, little is known regarding the mechanisms that may contribute to the rise of PRL. We measured serum PRL levels in 54 HIV-infected and 85 healthy age-matched men. The association between PRL levels among anti-PRL autoantibodies and other clinical variables in HIV-infected men was studied. We also evaluated the changes in serum PRL levels by chromatographic separation (affinity with protein G and gel filtration) after a 10-mg iv bolus of metoclopramide. HIV-infected men had higher serum PRL levels compared with healthy men. Sera from 9 of the 54 (16.7%)HIV-infected men were found to have hyperprolactinemia. Moreover, the anti-PRL autoantibody was present in four of nine (44.4%)HIV-infected men with hyperprolactinemia; it was also associated with hyperprolactinemic status. Serum total PRL levels were higher in HIV-infected men with anti-PRL autoantibodies than hyperprolactinemic HIV-infected men without anti-PRL autoantibodies; by contrast, free PRL levels were lower. In HIV-infected men with anti-PRL autoantibodies, gel filtration showed that big big PRL isoform was present as the predominant circulating form of PRL throughout each measurement after iv metoclopramide. By contrast, the predominant isoform of PRL in serum from healthy men and HIV-infected men who were anti-PRL autoantibody negative was little PRL. On the other hand, high serum total PRL levels were observed at each measurement throughout the metoclopramide test in HIV-infected men with anti-PRL autoantibodies; however, the serum free PRL levels were similar to those found in subjects without anti-PRL autoantibodies. These data demonstrated that anti-PRL autoantibodies are associated with hyperprolactinemic status in HIV-infected subjects, particularly in those with high serum PRL levels.

Adult↗
PubMed2002

[Employee assistance program].

Recently the EAP has received much attention in Japan. The first EAP service in the US was conducted by employees who had recovered from alcohol problems. In the early days EAP providers focused on addiction, but mainly after 1980 they expanded their service areas to include mental health, marital problems, legal problems and financial problems. In Japan the EAP was first received attention as a counseling resource outside the workplace where employees could seek professional help confidentially, but the main reasons why this system now interests employers are as a risk-management tool and an outsourcing of mental health services, since the growing number of mental health cases in the workplace has been a big issue for employers. Two movements have also contributed to more recognition of the EAP: one is guidelines on compensation for mental health cases in the workplace and the other is guidelines on mental health promotion in the workplace. There are four types of EAP systems: internal EAP, external EAP, combination EAP, and consortium EAP. EAP core technology consists of 8 functions including problem identification, Crisis intervention, Short-term intervention, Consultation with work organization leader. The literature on cost-benefit analysis of the EAP is very limited. Although the available data suggest that the EAP is highly cost-effective, further studies are needed with the sufficient statistical quality. In Japan the most important issues in the EAP are the standardization and quality assurance of EAP services. For this purpose development of a good educational system for EAP professionals is needed.

Cost-Benefit Analysis↗
PubMed1978

[Late results of the interintestinal anastomosis condition after congenital obstruction of the small intestine].

The data concerning the late results of resection of the small intestine carried out for congenital small intestine ileus in 43 newborns are set forth. The results of an experimental study on 75 puppies subjected to resection of the small intestine with application of various anastomoses are covered in the article. The evaluation of late results suggests that end-to-end anastomosis is the best one; end-to-side anastomosis gives fair results, while side-to-side anastomosis is the least physiological one as it results in the formation of big "blind ducts", and the longlasting stasis of intestinal contents in these "blind ducts" leads to the development of various complication. The children, who underwent the small intestine resection, should be under dispensary and periodic clinical observation.

Adolescent↗
PubMed1995

Software package for the management of sequencing projects using lacI transgenic animals.

The bacterial lacI gene has been used for many years as a mutational target for the study of the mechanisms of mutation. A wealth of information has been collected for many mutagenic treatments and in strains with diverse DNA repair backgrounds. Recently this gene has been used in the construction of a transgenic mouse, named Big Blue, and a transgenic rat, as well as a rat cell line. The lacI gene in these animals and cells can conveniently be recovered and analyzed in bacteria. This makes it possible to study mutagenic potential of chemical compounds in vivo using a mammal. Tissue, strain, and gender specificity can be addressed. In addition, mutations recovered from tumour tissues or from animals with specific genetic backgrounds can be analyzed conveniently. The mammalian systems can produce large numbers of mutants that require computer assistance to manage the samples and the resulting DNA sequence data. Accordingly, a computer software system was developed. The system maintains an inventory of bacteriophage lambda lacI mutants and allows entry of mutant sequences while performing accuracy checks on the data. The software features several options for displaying lists of mutants. The system can perform several analyses, including mutant class compilations, mutational spectra comparisons, and clonal expansions analysis. An extensive database obtained from the bacterial lacI system is included with the software and can be analyzed along with mutants derived from transgenic animals.

Algorithms↗
PubMed1990

Effect of endothelins on cytosolic free calcium concentration in neuroblastoma NG108-15 and NCB-20 cells.

The effects of endothelin(ET)-1, ET-2, ET-3 and Big ET on intracellular level of Ca2+ ([Ca2+]i) were studied in neuroblastoma NG108-15 and NCB-20 cells. All ETs, except Big ET, induced an increase in [CA2+]i in NG108-15 cells in a dose-dependent manner, with EC50: 6.7, 11.2 and 71 nM, respectively. However, none of the ET increased [Ca2+]i in NCB-20 cells. Calcium channel blockers diltiazem or nicardipine had no effect on ET-induced increase in [CA2+]i, but extracellular Ca2(+)-depletion significantly reduced the response of NG108-15 cells to ETs. NG108-15 cells exhibited a homologous desensitization to sequential addition of ETs, but no heterologous desensitization among ET, bradykinin and PAF was observed. These data suggest that ET-induced receptor activation results in increased intracellular Ca2+ via a non voltage calcium channel mechanism and intracellular Ca2+ release.

Animals↗
PubMed2002

Aggregation of gramicidin A in phospholipid Langmuir-Blodgett monolayers.

The aggregation of Gramicidin A (gA) in dipalmitoylphosphatidylcoline (DPPC) monolayers is investigated by both thermodynamic and structural methods. Compression isotherm analysis and atomic force microscopy (AFM) observations are performed. Our experimental results indicate that gA aggregation does occur in DPPC monolayers even at very low gA concentration (about 8 x 10(-4) mol%). At the low gA concentration limit, the aggregation process seems to be mainly horizontal (i.e., side-by-side, into the monolayer plane), following a fractal pattern growth producing the formation of typical, flat (0.5 nm height) "doughnut" structures, with a diameter of approximately 150 nm. These structures appear to be composed of smaller subunits (about 70 nm diameter) showing the same doughnut structure. At a molar fraction of approximately 3.8 mol%, the big doughnuts start to disaggregate and only small doughnuts appear. Above a gA concentration of approximately 4.4 mol%, all doughnuts (large and small) disappear, and the morphology assumes the appearance of a patchwork of two distinct phases: one that, being very flat, can be associated with a gA-free or gA-poor DPPC phase, and a second one, characterized by a more corrugated surface, associated with a gA-rich DPPC phase. At gA concentration of approximately 5 mol%, a percolation transition in the gA-rich DPPC phase occurs. Thermodynamic data indicate that the maximum of miscibility between gA and DPPC molecules occurs at approximately 28 mol%, suggesting that gA could aggregate in hexamers that are, on average, bound to 16 DPPC molecules. At the same concentration, AFM images show a network of small gA aggregation units of a size compatible with gA hexamers.

1,2-Dipalmitoylphosphatidylcholine↗
PubMed1993

Potent and selective inhibitors of an aspartyl protease-like endothelin converting enzyme identified in rat lung.

Two structurally distinct series of potent and selective inhibitors of an aspartyl protease-like endothelin converting enzyme (ECE) activity identified in the rat lung have been developed. Pepstatin A, which potently inhibits the rat lung ECE, served as the basis for the first series. Alternatively, selected renin inhibitors containing the dihydroxyethylene moiety were shown to be inhibitors of rat lung activity. Subsequent modifications improved inhibition of the rat lung ECE while eliminating renin activity. Both series of ECE inhibitors demonstrated a range of selectivity over Cathepsin D. Water-solubilizing moieties were appended onto selected compounds to facilitate in vivo testing. Partial reduction of the pressor response to exogenously administered Big ET-1 was observed with selected rat lung ECE inhibitors.

Amino Acid Sequence↗
PubMed2006

Deficiency in OGG1 protects against inflammation and mutagenic effects associated with H. pylori infection in mouse.

BACKGROUND: Helicobacter pylori infection is associated with gastric cancer. Study with the Big Blue mouse model has reported a mutagenic effect associated with the H. pylori infection, as a result in part of oxidative DNA damage. The present work investigates the consequences of a deficiency in the OGG1 DNA glycosylase, responsible for the excision of 8-oxo guanine, on the inflammatory and genotoxic host response to the infection. MATERIALS AND METHODS: Big Blue Ogg1-/- C57BL/6 mice were orally inoculated with H. pylori strain SS1 or vehicle only, and sacrificed after 1, 3, or 6 months. The serologic response, histologic lesions, mutant frequency, and spectra of mutations were assessed in the stomach and compared to what observed in the wild-type (Wt) context. RESULTS: Inflammatory lesions induced in the gastric mucosa of H. pylori-infected mice, corresponding to a moderate gastritis, were less severe in Ogg1-/- than in Wt Big Blue mice. Analysis of antimicrobial humoral immunity exhibited a lower IgG2a serum level (Th1 response) after 6 months of infection in Ogg1-/- than in the Wt mice. In these conditions, the H. pylori-SS1 infection in the Ogg1-/- mice did not induce a mutagenic effect at the gastric epithelial cells level, either after 3 or 6 months. CONCLUSIONS: The inactivation of the OGG1 DNA glycosylase in mouse leads to less severe inflammatory lesions and abolished the mutagenic effect at the gastric epithelial cells level, induced by the H. pylori infection. These data suggest for the OGG1deficiency a protective role against inflammation and genotoxicity associated to the H. pylori infection.

Animals↗
PubMed2004

Oral healthcare systems in the extended European union.

UNLABELLED: This article reports a survey of the systems for the provision of oral healthcare in the 28 member and accession states of the EU/EEA in 2003. Descriptions of the systems were collected from the principal dental advisers to governments in the individual states. In many states these were the Chief Dental Officers (CDOs). In states without a CDO, descriptions were gathered from CDO equivalents or senior academics. A template (model description) was used to guide all respondents. Additional statistical information on oral healthcare costs and workforce was collected from the Council of European Chief Dental Officers, WHO and World Bank websites. The study showed that in broad terms there were six patterns (Beveridgian, Bismarkian, The Eastern European (in transition), Nordic, Southern European and Hybrid) for the administration and financing of oral healthcare in the expanding EU. The extent and nature of government involvement in planning and coordinating oral healthcare services and the numbers and pay of the oral healthcare workforce varied between the different models. The biggest recent changes in European oral healthcare were found to have occurred in Eastern Europe, where there has been wide scale privatization of the previously public dental services. However, most of the EU accession (Eastern European) states seemed to be slowly developing insurance systems to cover oral health treatment costs. In the existing EU/EEA, the public dental services such as those in the Nordic countries still have strong political support and some expansion has occurred. In Southern Europe public dental services seemed to have gained some acceptance for the treatment of children and special needs groups. In UK, which has a unique public dental service system, there are plans to make big changes in the delivery, commissioning and remuneration of dental services in the near future. Some EU member states which operate the Bismarkian system with health insurances offering wide population coverage, comprehensive treatment and benefits connected with frequent dental visits, were reported to be experiencing financial problems. The study also indicated that at present, with the exception of Portugal and Spain, where there is dynamic growth in the numbers of dentists, the overall size of the EU/EEA oral health workforce is expanding fairly slowly. Only a minority of member states appeared to collect data on uptake of services and care costs and there were great difficulties in assessing outcomes of care. The data on costs appeared to show wide variations from member state to member state in per capita spending on oral healthcare. In the majority of states, however, costs, especially those in the private sector, could only be estimated. Nevertheless, at a 'macro' level, the study indicated that, in 2000, the 28 member and accession states of the EU/EEA had a total population of 456 million and an oral health workforce of 900,000 (some 300,000 of whom were dentists) and that the cost of oral healthcare was about EUR 54,000,000,000. CONCLUSION: The study showed wide variations in oral healthcare provision systems between EU/EEA member and accession states and no evidence of harmonization in the past.

Adult↗
PubMed1999

Vertebrate genome evolution: a slow shuffle or a big bang?

In vertebrates it is often found that if one considers a group of genes clustered on a certain chromosome, then the homologues of those genes often form another cluster on a different chromosome. There are four explanations, not necessarily mutually exclusive, to explain how such homologous clusters appeared. Homologous clusters are expected at a low probability even if genes are distributed at random. The duplication of a subset of the genome might create homologous clusters, as would a duplication of the entire genome. Alternatively, it may be adaptive for certain combinations of genes to cluster, although clearly the genes must have duplicated prior to rearrangement into clusters. Molecular phylogenetics provides a means to examine the origins of homologous clusters, although it is difficult to discriminate between the different explanations using current data. However, with more extensive sequencing and mapping of vertebrate genomes, especially those of the early diverging chordates, it should soon become possible to resolve the origins of homologous clusters.

Animals↗
PubMedDate not supplied

Health care for children and youth in the United States: annual report on patterns of coverage, utilization, quality, and expenditures by income.

OBJECTIVES: To examine differences by income in insurance coverage, health care utilization, expenditures, and quality of care for children in the United States. METHODS: Two national health care databases serve as the sources of data for this report: the 2000-2002 Medical Expenditure Panel Survey (MEPS) and the 2001 Nationwide Inpatient Sample (NIS) from the Healthcare Cost and Utilization Project (HCUP). In the MEPS analyses, low income is defined as less than 200% of the federal poverty level and higher income is defined as 200% of the federal poverty level or more. For the HCUP analyses, median household income for the patient's zip code of residence is used to assign community-level income to individual hospitalizations. RESULTS: Coverage. Children from low-income families were more likely than children from middle-high-income families to be uninsured (13.0% vs 5.8%) or covered by public insurance (50.8% vs 7.3%), and less likely to be privately insured (36.2% vs 87.0%). Utilization. Children from low-income families were less likely to have had a medical office visit or a dental visit than children from middle-high-income families (63.7% vs 76.5% for office-based visits and 28.8% vs 51.4% for dental visits) and less likely to have medicines prescribed (45.1% vs 56.4%) or have utilized hospital outpatient services (5.2% vs 7.0%), but more likely to have made trips to the emergency department (14.6% vs 11.4%). Although low-income children comprise almost 40% of the child population, one quarter of total medical expenditures were for these children. Hospital Discharges. Significant differences by community-level income occurred in specific characteristics of hospitalizations, including admissions through the emergency department, expected payer, mean total charges per day, and reasons for hospital admission. Leading reasons for admission varied by income within and across age groups. Quality. Low-income children were more likely than middle-high-income children to have their parents report a big problem getting necessary care (2.4% vs 1.0%) and getting a referral to a specialist (11.5% vs 5.3%). Low-income children were at least twice as likely as middle-high-income children to have their parents report that health providers never/sometimes listened carefully to them (10.0% vs 5.1%), explained things clearly to the parents (9.6% vs 3.4%), and showed respect for what the parents had to say (9.2% vs 4.2%). Children from families with lower community-level incomes were more likely to experience ambulatory-sensitive hospitalizations. Racial/Ethnic Differences Between Income Groups. Use and expenditure patterns for most services were not significantly different between low- and middle-high-income black children and were lower than those for white children. CONCLUSIONS: While health insurance coverage is still an important factor in obtaining health care, the data suggest that efforts beyond coverage may be needed to improve access and quality for low-income children overall and for children who are racial and ethnic minorities, regardless of income.

Adolescent↗
PubMed2005

Retroperitoneal germ cell tumors: a clinical study of 12 patients.

PURPOSE: The aim of the study was to examine the clinical presentation, method(s) of treatment, complications, and results in newborns and infants with retroperitoneal germ cell tumors (GCTs). METHODS: A retrospective chart review of all patients treated between 1974 and 2002 for GCT located in the retroperitoneum in 2 institutions identified 12 patients with histologically proven retroperitoneal GCT. Vital data concerning pregnancy and delivery were analyzed. Age at diagnosis and symptoms were recorded, as well as possibly associated anomalies. Data concerning surgical treatment, perioperative and postoperative complications, histological staging, and final outcome were all analyzed. RESULTS: In 3 patients, the diagnosis had been made antenatally between 31 and 35 weeks of gestation. In 1 patient, the diagnosis was made at birth, and in 8 later in life (ages 3, 5, 7, 8, 8, 11, 18, and 24 months). Symptoms in these 8 boys and 4 girls were abdominal distension and a palpable upper abdominal mass, right-sided in 5, left-sided in 5, and central in 2; the tumor was usually big. Associated anomalies were noted in 4 patients and were chromosomal in 3 (Down syndrome in 2 and Klinefelter syndrome in 1). One baby died of uncontrollable bleeding during an emergency operation immediately after traumatic birth. The other 11 infants survived. Four other patients showed serious perioperative complications (1 caval vein tear, 1 choledochal tear, 1 cyst rupture, and 1 esophagogastric tear) which were managed without further consequences. Histologically, 4 tumors were mature teratomas, 6 were immature teratomas (grade I in 4, grade II in 1, and grade II-III in 1), and 2 were malignant yolk sac tumors (YSTs). The patients with YSTs underwent surgical biopsy, followed by chemotherapy and excision of the remaining tumor and of the metastases. No adjuvant treatment was administered in the patients with benign disease. Nine survivors with benign tumor are disease-free between 1 and 30 years after surgery. Two patients with YST have now been in remission for 6 and 5 years, respectively. CONCLUSIONS: Both this study and the literature review performed testify to the extreme rarity of GCTs in the retroperitoneum. Surgical removal of the tumors appeared to be hazardous because of the extent of the tumor, the displacement and elongation of adjacent structures and organs, and/or the adhesion of the tumor to surrounding tissues; this resulted in several perioperative complications. The long-term results are good, however, with 9 of 10 patients with benign tumors in good health after a mean follow-up of 12 years, and with the 2 patients with YST in remission for 6 and 5 years, respectively.

Abnormalities, Multiple↗