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Biomedical subjects

P Ferreira

Publications and source records attributed to P Ferreira.

At least 19 recordsLinked to original sources

Identification of seven novel germline mutations in the human E-cadherin (CDH1) gene.

Hereditary diffuse gastric cancer (HDGC) is a cancer predisposition syndrome caused by germline mutation of the gene encoding the tumour-suppressor E-cadherin (CDH1). We describe the search for CDH1 mutations in 36 new diffuse gastric cancer families. All 16 CDH1 exons, neighbouring intronic sequence and an essential promoter region were screened by DNA sequencing. We detected nine different mutations, seven of which were novel. Of the seven novel mutations, five were identified in families who met the IGCLC clinical criteria for HDGC. Two mutations resulted in a premature stop codon and truncation of the protein. Three mutations affected splice sites; two of the splice-site mutations were shown by RT-PCR to disturb normal CDH1 splicing, while the third splice-site mutation was present in two unrelated HDGC families. The remaining two mutations resulted in amino acid substitutions and impaired the ability of E-cadherin protein to form cellular aggregates and suppress invasion in vitro. Together with the occurrence of extra-gastric tumours such as lobular breast and colorectal cancer, these findings further extend the types of CDH1 mutations and the spectrum of tumours associated with HDGC.

Adult↗

Release of ferulic acid and feruloylated oligosaccharides from sugar beet pulp by Streptomyces tendae.

Given several promising industrial applications of ferulic acid, this study was designed to identify actinomycete strains able to release high levels of this acid from sugar beet pulp (SBP). Out of 47 strains tested, 37% were found to release free ferulic acid from the growth substrate. One strain, identified as Streptomyces tendae by 16S RNA gene sequencing, was capable of releasing 80% of the ferulic acid ester-linked to the pectin in SBP after 5 days of growth. These data suggest that some actinomycetes are able to release ferulic acid and feruloylated oligosaccharides from SBP. During growth on SBP, it seems that Streptomyces species solubilize and release feruloylated oligosaccharides by specific carbohydrase activities before de-esterification and release of free ferulic acid.

Agriculture↗

Modification of the biopolymer castor oil with free isocyanate groups to be applied as bioadhesive.

Surgical adhesives have been used for several applications, including haemostasis, sealing air leakages and tissue adhesion. The aim of this work was to develop a biodegradable urethane-based bioadhesive containing free isocyanate groups. This material presents the advantage of being biodegradable, biocompatible and having the capacity of reacting with amino groups present in the biological molecules. A urethane based on castor oil (CO) was synthesized by reaction of the molecule with isophorone diisocyanate (IPD). The characterization of the material was accomplished by different techniques: ATR-FT-IR (attenuated transmittance reflection-Fourier transform infrared), swelling capacity determination, evaluation of the moisture curing kinetics, reaction with aminated substrates and determination of surface energy by contact angle measurement. The study of the urethane thermal properties was performed by DMTA (dynamical mechanical thermal analysis) and TGA (thermogravimetric analysis). The haemocompatibility of the urethane was also evaluated by thrombosis and haemolysis tests.

Biopolymers↗

Synthesis and characterization of membranes obtained by graft copolymerization of 2-hydroxyethyl methacrylate and acrylic acid onto chitosan.

Chitosan based membranes to be applied on wound healing as topical drug delivery systems were developed by graft copolymerization of acrylic acid (AA) and 2-hydroxyethyl methacrylate (HEMA) onto chitosan using cerium ammonium nitrate as chemical initiator. Evidence for graft copolymerization of the vinyl monomers onto chitosan was obtained by FTIR and DMTA. Swelling degree, cytotoxicity, thrombogenicity and haemolytic activity of these membranes were evaluated. Chitosan-graft-AA-graft-HEMA showed to be the best matrix for drug delivery systems than chitosan-graft-AA because it retains good swelling properties, but the content in HEMA has improved cytocompatibility, hemocompatibility and thrombogenic character.

Acrylates↗

Mutation of DNAJC19, a human homologue of yeast inner mitochondrial membrane co-chaperones, causes DCMA syndrome, a novel autosomal recessive Barth syndrome-like condition.

BACKGROUND: A novel autosomal recessive condition, dilated cardiomyopathy with ataxia (DCMA) syndrome, has been identified in the Canadian Dariusleut Hutterite population, characterised by early onset dilated cardiomyopathy with conduction defects, non-progressive cerebellar ataxia, testicular dysgenesis, growth failure, and 3-methylglutaconic aciduria. OBJECTIVE: To map DCMA syndrome and identify the mutation underlying this condition. METHODS: A genome wide scan was undertaken on consanguineous Hutterite families using a homozygosity mapping approach in order to identify the DCMA associated chromosomal region. Mutation analysis was carried out on positional candidate genes in this region by sequencing. Reverse transcriptase polymerase chain reaction and bioinformatics analyses were then used to characterise the mutation and determine its effect on the protein product. RESULTS: The association of DCMA syndrome with a 2.2 Mb region of chromosome 3q26.33 was found. A disease associated mutation was identified: IVS3-1 G-->C in the DNAJC19 gene, encoding a DNAJ domain containing protein of previously unknown function (Entrez Gene ID 131118). CONCLUSIONS: The DNAJC19 protein was previously localised to the mitochondria in cardiac myocytes, and shares sequence and organisational similarity with proteins from several species including two yeast mitochondrial inner membrane proteins, Mdj2p and Tim14. Tim14 is a component of the yeast inner mitochondrial membrane presequence translocase, suggesting that the unique phenotype of DCMA may be the result of defective mitochondrial protein import. It is only the second human disorder caused by defects in this pathway that has been identified.

Abnormalities, Multiple↗

Correction of long term joint contractures of the hand by distraction. A case report.

Joint contractures are a common complication of hand trauma. The conventional treatment consists of arthrolysis, tenolysis and occasionally arthrodesis. Frequently, this does not achieve a good result, particularly when there has been a long delay in presentation. Progressive lengthening of a joint by distraction (joint distraction) allows the release of joint contractures even in cases of failure of traditional methods. We present a case of a delayed (20 years) work related traumatic flexion deformity of the PIP joint of the left index and middle fingers. This was the result of a complete division of both flexor tendons of both fingers. The range of movements, both active and passive, was limited to 90/100 degrees in the index finger and 95/100 degrees in the middle finger. Following joint distraction using our lengthening device (Antãotrade mark, Portugal) the patient was able to achieve an active and passive range of movements of 10/100 degrees for the PIP joint of the index finger and 40/100 degrees of the middle. This clinical case shows the simplicity and application of our technique for the correction of joint contractures.

Adult↗

Germline E-cadherin mutations in hereditary diffuse gastric cancer: assessment of 42 new families and review of genetic screening criteria.

BACKGROUND: Mutations in the E-cadherin (CDH1) gene are a well documented cause of hereditary diffuse gastric cancer (HDGC). Development of evidence based guidelines for CDH1 screening for HDGC have been complicated by its rarity, variable penetrance, and lack of founder mutations. METHODS: Forty three new gastric cancer (GC) families were ascertained from multiple sources. In 42 of these families at least one gastric cancer was pathologically confirmed to be a diffuse gastric cancer (DGC); the other family had intestinal type gastric cancers. Screening of the entire coding region of the CDH1 gene and all intron/exon boundaries was performed by bi-directional sequencing. RESULTS: Novel mutations were found in 13 of the 42 DGC families (31% overall). Twelve of these mutations occur among the 25 families with multiple cases of gastric cancer and with pathologic confirmation of diffuse gastric cancer phenotype in at least one individual under the age of 50 years. The mutations found include small insertions and deletions, splice site mutations, and three non-conservative amino acid substitutions (A298T, W409R, and R732Q). All three missense mutations conferred loss of E-cadherin function in in vitro assays. Multiple cases of breast cancers including pathologically confirmed lobular breast cancers were observed both in mutation positive and negative families. CONCLUSION: Germline truncating CDH1 mutations are found in 48% of families with multiple cases of gastric cancer and at least one documented case of DGC in an individual under 50 years of age. We recommend that these criteria be used for selecting families for CDH1 mutational analysis.

Adolescent↗

Therapeutic vaccine against Streptococcus sobrinus-induced caries.

Streptococcus sobrinus produces a virulence-associated immunomodulatory protein (VIP) which suppresses the host-specific immune response and induces the early production of IL-10. In this study, we evaluated the effects of therapeutic immunization with this VIP on the incidence of caries in S. sobrinus-infected rats. Groups of Wistar rats were orally infected with S. sobrinus and fed with sucrose-sweetened drinking water ad libitum. Five days later, rats were immunized intranasally with active or heat-inactivated VIP plus alum as adjuvant or PBS plus adjuvant (sham-immunized). After 3 wks, all rats were re-immunized as above. Evaluation of dental caries showed that VIP-immunized animals had significantly fewer enamel sulcal and proximal caries lesions than did the sham-immunized animals (p < 0.001). The protective effects following therapeutic VIP immunization were attributed to the induced salivary immunoglobulin A specific to the VIP. These results offer a promising and safe strategy for the development of a vaccine against dental caries.

Analysis of Variance↗

RPGRIP1s with distinct neuronal localization and biochemical properties associate selectively with RanBP2 in amacrine neurons.

RPGR and RPGRIP1 are molecular partners with vital roles in retinal function. Mutations in RPGR are implicated in heterogeneous retinal phenotypes, while those in RPGRIP1 lead to Leber congenital amaurosis. RPGR and RPGRIP1s differentially localize in photoreceptors among species. This may contribute to phenotype disparities among species bearing mutations in RPGR. However, it cannot account for the phenotype heterogeneity associated with RPGR- and RPGRIP1-linked mutations in the human. The existence of RPGRIP1 isoforms with distinct cellular, subcellular localizations and biochemical properties in the retina is shown. High mass RPGRIP1 isoforms, p175/p150, enriched in the outer segment (OS) compartment of photoreceptors are identified. The remaining isoforms are present across subcellular fractions, including nuclei and are soluble. The p175/p150 are predominantly sequestered in the cytoskeleton-insoluble fraction of OS and nuclei. In selective amacrine cells, and in the transformed photoreceptor line, 661W, RPGRIP1s localize at restricted foci to nuclear pore complexes and/or the vicinity of these. Among the nucleoporins, RPGRIP1 isoforms selectively associate in vivo with RanBP2 (Nup358). RPGRIP1s also decorate microtubules in 661W cells and occasionally form coiled-like inclusion bodies in the perikarya. These results support distinct but complementary functions of RPGRIP1 isoforms in cytoskeletal-mediated processes in photoreceptors and amacrine neurons, and may explain the Leber phenotype linked to RPGRIP1 mutations in humans. Moreover, the data implicate a role of RanBP2 in the pathogenesis of neuro(retino)pathies and as a docking station to mediate the nucleocytoplasmic shuttling of RPGRIP1s and their interaction with other partners in amacrine and 661W neurons.

Amacrine Cells↗

[Other indications for surfactant].

OBJECTIVE: The introduction of surfactant replacement therapy in the management of respiratory distress syndrome in the premature infant was a remarkable advance in neonatal intensive care. In the last few years, recognition of the role played by surfactant inactivation in the pathogenesis of other respiratory diseases of the newborn has justified new therapeutic applications. The aim of this study was to evaluate the efficacy of treatment with natural exogenous surfactant in situations with secondary surfactant deficiency. METHODS: We performed a retrospective analysis of the evolution of 15 newborn infants treated with natural exogenous surfactant. Nine infants had meconium aspiration syndrome, five had congenital pneumonia and one had adult respiratory distress syndrome. Oxygenation indexes before and after surfactant treatment as well as clinical and radiographic evolution were compared. RESULTS: The 12 surviving infants showed improvement in oxygenation and radiographic alterations after surfactant administration. Three patients with meconium aspiration syndrome died. CONCLUSIONS: These results support the therapeutic use of exogenous surfactant in severe respiratory diseases of the newborn causing secondary surfactant deficiency.

Female↗

[Birdshot retinochoroidopathy. Five clinical cases analysis].

PURPOSE: The Birdshot retinochoroidopathy is a rare chronical intraocular inflammatory disease. The authors made a comparative study of the disease's clinical manifestations and its response to therapy. METHODS: We selected five patients who had ocular inflammation which presented on the ocular fundus examination typical multifocal yellow-white patches of depigmentation of choroid and pigment epithelium and vitreous inflammation. RESULTS: All the patients showed early spots of hypofluorescence that became hyperfluorescent in the venous phase of angiography, a depression of b-wave amplitude of electroretinogram, a blue-yellow defect of colour vision and carried HLA-A 29 antigen. We recorded an improvement of the visual acuity and of the clinical picture after treatment with corticosteroid and cyclosporine A. CONCLUSION: Even though Birdshot retinochoroidopathy is a rare pathology in Southern Europe, the cases presented allowed us to conclude that early diagnosis and appropriate therapy greatly improve the well being of these patients (Arch Soc Esp Oftalmol 2002; 77: 247-256).

Adult↗

Bidentate Lewis base adducts of methyltrioxorhenium(VII) and their application in catalytic epoxidation.

Methyltrioxorhenium(VII) (MTO) forms octahedral adducts with bidentate Lewis bases. These complexes were isolated and fully characterized, including X-ray crystallography. The compounds display distorted octahedral geometry in the solid state with a tendency of disorder concerning the Re central atom. At elevated temperatures, they undergo rapid ligand-exchange reactions in solution. The ease of this ligand exchange depends mainly on the Lewis basicity of the ligand. The more Lewis basic the ligand is, the stronger the metal-ligand interaction is, as can be shown by NMR spectroscopy. All examined complexes are temperature stable but quite sensitive to light and moisture. In the presence of H(2)O(2), the complexes form very active and highly selective epoxidation catalysts. Peroxo complexes are generated, and at least one of the Re-N interactions is cleaved during this process. Total ligand dissociation only occurs in the case of very weakly coordinating bidentate ligands. The peroxo complexes of the MTO Lewis base adducts are, in general, more sensitive to water than MTO itself.

Journal Article↗

Novel microporous europium and terbium silicates.

The synthesis and structural characterization of the first examples of microporous europium(III) and terbium(III) silicates (Na(4)K(2)X(2)Si(16)O(38) x 10H(2)O, X = Eu, Tb) are reported. The structure of these solids was solved by powder X-ray diffraction ab initio (direct) methods and further characterized by chemical analysis (EDS), thermogravimetric analysis (TGA), scanning electron microscopy (SEM), (23)Na and (29)Si magic-angle spinning (MAS) NMR, and luminescence spectroscopy. Both materials display interesting photoluminescence properties and present potential for applications in optoelectronics. This work illustrates the possibility of combining in a given silicate microporosity and optical activity.

Journal Article↗

Heterologous expression and characterization of a rare Gaucher disease mutation (c.481C > T) from a Canadian aboriginal population using archival tissue samples.

Gaucher disease is an inherited sphingolipidosis resulting from deleterious mutations in the glucocerebrosidase gene. Through direct sequence analysis of genomic DNA from whole blood, fibroblast cultures, and formalin-fixed archival tissue samples, we have identified a rare homozygous C > T transition at cDNA nucleotide 481 of the glucocerebrosidase gene that results in a proline to serine amino acid substitution (p.P122S) in an aboriginal family of Cree descent in northern Alberta, Canada. A 13-month-old boy (JB) presented with severe visceral Gaucher disease and was treated with enzyme replacement. Currently, at 11 years he is developmentally delayed, with oculomotor apraxia. A cousin (MS) had previously died at age 7 from complications of severe Gaucher disease, before enzyme replacement therapy was available. She was also developmentally delayed. Heterologous expression of this allele using a baculovirus expression system revealed 19.2% of normal enzyme activity on the artificial substrate 4-methylumbelliferyl beta-d-glucopyranoside (4MUGP). Genotype/phenotype correlation is complicated by incomplete clinical details, enzyme replacement therapy, and the difficulty in excluding other genetic and environmental causes of developmental delay. However the development of oculomotor apraxia in JB suggests a Type 3 Gaucher phenotype. The only previous report of this mutation was also from a member of the Cree Nation, who has had a rather similar clinical course. A protocol is described for the isolation of genomic DNA from formalin-fixed bone marrow aspirate archival specimens obtained from the deceased for subsequent PCR-based sequence analysis and mutation detection. This technique will be applicable to the screening of this and other populations for the frequency of known Gaucher mutations where traditional DNA sources are unavailable.

Adolescent↗

Plasmapheresis as an effective treatment for opsoclonus-myoclonus syndrome.

A 6-year-old female diagnosed with idiopathic opsoclonus-myoclonus syndrome at 22 months of age who failed to respond to treatment with adrenocorticotropic hormone (ACTH), IV gammaglobulin (IVIG), and azathioprine is presented. Because of marked and progressive deterioration in motor function and speech, this patient received a course of plasmapheresis with concomitant steroids and azathioprine. Within 1 week, marked improvements in motor function were noted. Eighteen months later, the patient ambulates, walks without support, and attends a regular school in the appropriate grade level.

Azathioprine↗