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Quasispecies nature of three maize streak virus isolates obtained through different modes of selection from a population used to assess response to infection of maize cultivars.

Three maize streak virus (MSV) isolates were derived from an MSV population used to assess the response to infection of maize cultivars. Isolate SP1 was obtained from this population through short acquisition and inoculation periods (1 and 5 min, respectively), using a single Cicadulina mbila vector. Isolate SP2 was derived from SP1 after transmission to a wild perennial host (Coix lacryma-jobi), on which it was maintained for about 4 years without insect transmission. Isolate N2A, the most pathogenic isolate, was obtained from the initial population after serial passages on almost completely resistant inbred maize lines. The complexity of each isolate was analysed by RFLP analysis and sequencing based on 120 SP1 clones, 36 SP2 clones and 40 N2A clones. All three isolates were composed of different but related clones, consistent with a quasispecies structure. The mutations were distributed throughout the genome. Mutation frequencies, based on all available sequences, were 3.8 x 10(-4) for SP1, 10.5 x 10(-4) for SP2 and 6.9 x 10(-4) for N2A. As expected from the bottleneck selection step, the intra-isolate variability of SP1 was relatively low. Comparison between SP1 and SP2 showed that SP1 heterogeneity increased during maintenance on the wild host. Furthermore, the consensus sequences of SP1 and SP2 differed by two non-synonymous substitutions in the complementary sense gene repA. N2A had a relatively low degree of heterogeneity, but was composed of several sub-populations. The results reflect the influence of the mode of selection of MSV isolates on their quasispecies organization, i.e. distribution of variants, and master sequence.

Base Sequence↗

Molecular epidemiology of a large outbreak of hepatitis B linked to autohaemotherapy.

BACKGROUND: Unregulated skin-piercing procedures potentially facilitate the transmission of bloodborne pathogens. In February, 1998, a patient who had recently received autohaemotherapy at an alternative medicine clinic in the UK was diagnosed with acute hepatitis B. The autohaemotherapy procedure involved the drawing of 1 mL of the patient's blood, mixing with saline, and reinjection of the autologous blood mixture. We investigated the extent of hepatitis B virus (HBV) infection in patients and staff of the clinic. METHODS: Patients who had attended the clinic between January, 1997, and February, 1998, were tested for serological markers of HBV, and for HBV DNA by PCR. HBV DNA was sequenced to assess the relatedness of the virus identified in the cases. We analysed the number and dates of visits with regard to HBV status. FINDINGS: Serum samples were received from 352 patients and four staff members. Serological evidence of exposure to HBV was found in 57 (16%). Of the 33 patients and staff who were positive for hepatitis B surface antigen, 30 (91%) showed complete nucleotide identity in the DNA segments derived from the surface and core genes. Five patients with linked infection had markers of chronic hepatitis B, and one of these was regarded as the likely source of the outbreak. The attack rate was associated with the number of visits (p<0.0001) and the week of visit (p=0.011). Contaminated saline in a repeatedly used bottle was the probable vehicle of transmission. INTERPRETATION: We have described a large community-based outbreak of hepatitis B due to transmission by a single HBV variant. Our findings emphasise the continuing risk of transmission of bloodborne viruses in all health-care settings where skin-piercing procedures are used.

Adult↗

Central nervous system infections due to Abiotrophia and Granulicatella species: an emerging challenge?

Although Abiotrophia and Granulicatella species, previously referred to as nutritionally variant streptococci, were initially identified over 40 years ago, isolation of these pathogens from the central nervous system (CNS) was first noted only recently. Recognition of CNS involvement with these organisms is of great concern given the association of Abiotrophia/Granulicatella infections with increased morbidity and mortality as well as greater bacteriologic failure and relapse rates. We describe A. defectiva and G. adiacens CNS infections in two patients and review the existing literature of CNS involvement with these bacteria. The clinical presentation and initial cerebrospinal fluid analysis has varied substantially across reported patients. While most infections have been characterized primarily by a localized infection (e.g., abscess), evidence of meningitis has usually also been present. Furthermore, nearly all cases have followed neurosurgical procedures suggesting possible introduction of the organism into the CNS at the time of surgery. Given the significant negative clinical impact of Abiotrophia/Granulicatella infections, elucidation of the emerging epidemiology of CNS infections with these bacteria is warranted.

Adult↗

Pathogenicity and comparative evolution in vivo of the transitional quasispecies SIVsmmPBj8.

During 14 months of infection of a pig-tailed macaque, the acutely lethal simian immunodeficiency virus SIVsmmPBj14 (SIV-PBj14) evolved from the minimally pathogenic strain SIVsmm9. The virus isolated at 8 months (SIV-PBj8) exhibited properties of both SIVsmm9 and SIV-PBj14, indicating that a phenotypic transition occurred between 6 and 10 months. To assess the influence that this new composition of biologic properties might have on pathogenicity, three pig-tailed macaques were inoculated intravenously with SIV-PBj8. Although no animals developed the severe acute disease syndrome typical of SIV-PBj14, all had high levels of viremia and died of AIDS at 4, 10. 5, and 32 months. Characterization of the SIV-PBj8-derived quasispecies that evolved in these macaques showed that at 4 days after inoculation, viruses from all three animals exhibited in vitro properties different from those of the inoculum. By 4 months, the initial phenotypic profiles had changed, with the quasispecies in plasma from the animal (J90232) that died at this time most closely resembling SIV-PBj14, not SIV-PBj8. Phylogenetic trees of the gp41/Nef region of viruses in 4-month plasma from J90232 revealed three distinct populations with high bootstrap values: one group branched with SIVsmm9, one with SIV-PBj14, and one with SIV-PBj8 (ratio of clones, 5:9:5). Nucleotide sequence analysis suggested that some members of the original SIV-PBj8 quasispecies may have been evolving toward a SIV-PBj14-like genotype at the time macaque J90232 died. The use of SIV-PBj8, which was more pathogenic than SIVsmm9, but less pathogenic than SIV-PBj14, may provide the optimal genetic background on which to identify the minimal, multigenic determinants of the SIV-PBj14 phenotype. The results of our studies on SIV-PBj14 indicate that in some, but not all, cases of primate lentivirus infection more pathogenic variants evolve, selectively proliferate, and more than likely contribute to disease progression.

Animals↗

Short- and long-term clinical outcomes in rhesus monkeys inoculated with a highly pathogenic chimeric simian/human immunodeficiency virus.

A highly pathogenic simian/human immunodeficiency virus (SHIV), SHIV(DH12R), isolated from a rhesus macaque that had been treated with anti-human CD8 monoclonal antibody at the time of primary infection with the nonpathogenic, molecularly cloned SHIV(DH12), induced marked and rapid CD4(+) T cell loss in all rhesus macaques intravenously inoculated with 1.0 50% tissue culture infective dose (TCID(50)) to 4.1 x 10(5) TCID(50)s of virus. Animals inoculated with 650 TCID(50)s of SHIV(DH12R) or more experienced irreversible CD4(+) T lymphocyte depletion and developed clinical disease requiring euthanasia between weeks 12 and 23 postinfection. In contrast, the CD4(+) T-cell numbers in four of five monkeys receiving 25 TCID(50)s of SHIV(DH12R) or less stabilized at low levels, and these surviving animals produced antibodies capable of neutralizing SHIV(DH12R). In the fifth monkey, no recovery from the CD4(+) T cell decline occurred, and the animal had to be euthanized. Viral RNA levels, subsequent to the initial peak of infection but not at peak viremia, correlated with the virus inoculum size and the eventual clinical course. Both initial infection rate constants, k, and decay constants, d, were determined, but only the latter were statistically correlated to clinical outcome. The attenuating effects of reduced inoculum size were also observed when virus was inoculated by the mucosal route. Because the uncloned SHIV(DH12R) stock possessed the genetic properties of a lentivirus quasispecies, we were able to assess the evolution of the input virus swarm in animals surviving the acute infection by monitoring the emergence of neutralization escape viral variants.

Amino Acid Substitution↗

Genetic study of von Willebrand factor antigen levels &#x2264; 50 IU/dL identifies variants associated with increased risk of von Willebrand disease and bleeding.

BACKGROUND: von Willebrand disease (VWD) is a common inherited bleeding disorder caused by low levels or activity of circulating von Willebrand factor (VWF). Genetic susceptibility to VWF antigen (VWF:Ag) below normal (&#x2264; 50 IU/dL) in the general population is underexplored. OBJECTIVES: To identify genetic variants influencing VWF:Ag levels &#x2264; 50 IU/dL. METHODS: We performed a genome-wide association study in 926 cases with VWF:Ag levels &#x2264; 50 IU/dL and 12 846 controls from 7 studies from the Trans-Omics for Precision Medicine program. We then examined whether significant genome-wide findings were also associated with clinical diagnosis of VWD in 5 biobanks with 708 VWD cases and 1 286 069 controls, and with 6 bleeding and thrombotic disorders in FinnGen. RESULTS: Variants at 2 loci were associated (P < 5 &#xd7; 10-9) with VWF:Ag levels &#x2264; 50 IU/dL: ABO and VWF. The VWF index variant, p.Tyr1584Cys, is a rare (0.22%) missense variant with odds ratio (OR) of 78.58, while the ABO index variant is a common intronic variant with a smaller effect (OR = 2.52). Notably, both VWF (OR = 7.16) and ABO (OR = 1.57) variants were also associated (P < .025) with diagnosed VWD. Among p.Tyr1584Cys heterozygotes, the penetrance of VWF:Ag levels &#x2264; 50 IU/dL was 24.2% and the penetrance of diagnosed VWD was 0.3%. p.Tyr1584Cys was associated (P < .0042) with increased odds of heavy menstrual bleeding (OR = 1.27), iron deficiency anemia (OR = 1.55), and intrapartum hemorrhage (OR = 2.20), but decreased odds of deep vein thrombosis (OR = 0.54). CONCLUSIONS: Although there are currently conflicting interpretations of pathogenicity p.Tyr1584Cys, our results suggest that it is a low penetrance pathogenic variant that contributes to VWF:Ag levels &#x2264; 50 IU/dL, bleeding, and VWD.

Humans↗

Variants of the long control region of human papillomavirus type 16.

Expression of the human papillomavirus (HPV) E6 and E7 oncogenes is regulated on the transcriptional level by specific protein-binding sites contained in the viral long control region (LCR). Sequence changes within the LCR region may have an impact on the transcription of viral oncogenes, possibly resulting in differences in the oncogenic potential of the virus. The present study was designed to determine the sequence variability of the LCR of HPV 16 and to assess whether certain LCR variants do correlate with the clinical outcome of the disease of the uterine cervix. The entire LCR segment of HPV 16 was analysed from 37 cervical biopsy specimens derived from 28 women included in the Kuopio long-term prospective follow-up study. The LCR sequence was identical with the reference sequence in six HPV 16 isolates. Overall, 14 different HPV 16 LCR variants were identified. One of the variants showed sequence variation typical of the Asian-American variant lineage of HPV 16, and all the other variants appeared to belong to the European variant group. The European variants exhibited low genetic diversity, and only five of these LCR variants contained nucleotide changes involving known or proposed binding sites for transcription factors. The variants with changes at nucleotide positions 7193 and 7521 was the most prevalent, accounting for almost 37% of infections. This variant (7193; 7521) has been previously demonstrated to have similar transcriptional activity compared with the reference isolate by Veress and colleagues J Gen Virol 1999, 80, 1035-1043. The reference isolate, variant (7193; 7521) and variants with changes within transcription factor binding sites accounted for most of the infections, and no significant differences were found in the comparison of the distribution of these different LCR isolates in cases where the disease showed progression to severe cervical intraepithelial neoplasia (CIN) or carcinoma in situ (CIS). Notably, both the reference isolate and variant (7193; 7521) were also closely associated with infections showing more aggressive behaviour. According to the present findings, in European HPV 16 isolates, intratype genetic variation of the LCR region does not seem to be commonly responsible for differences in the pathogenicity of the virus and thereby for a risk of progressive infections.

DNA, Viral↗

Chemiluminescent analysis of Borrelia burgdorferi penicillin-binding proteins using ampicillin conjugated to digoxigenin.

Knowledge of the penicillin-binding proteins (PBPs) of Borrelia burgdorferi is important for understanding both the targets of beta-lactams used therapeutically for Lyme borreliosis and the complex membrane biology of the distinctive spirochetal pathogen which causes Lyme disease. In this study, the PBPs of a number of B. burgdorferi strains and variants were examined using a rapid and sensitive chemiluminescent assay which employs ampicillin conjugated to digoxigenin (dig-amp). The minimum inhibitory concentration of dig-amp for B. burgdorferi high-passage strain B31 (0.012 micrograms/ml) was essentially no different from that of free ampicillin (0.025 micrograms/ml). Dig-amp bound specifically to B. burgdorferi B31 PBPs with molecular masses of 92, 80, 65, 46, 40, 34, 31, 29, 22, 20 and 13 kDa; the 31 kDa and 34 kDa PBPs were proven to be OspA and OspB, respectively. All of the borrelial PBPs were present in the cytoplasmic membrane fraction of B. burgdorferi, findings consistent with their activities as PBPs but inconsistent with OspA and OspB as surface-exposed outer membrane lipoproteins. Furthermore, among the PBP profiles of other high- and low-passage variants of B. burgdorferi strains Sh-2-82, HB19, and N40, which differed somewhat from one another, OspD (28 kDa) but not OspC (22-25 kDa) also was strongly implicated as a PBP; however, OspC possessed a gel mobility easily misconstrued as that of a 26 kDa PBP often expressed reciprocally with OspB. The ramifications of classifying OspA, OspB, and OspD as PBPs are discussed. While the current inability to genetically manipulate B. burgdorferi hinders determining which of the borrelial PBPs are essential for spirochetal viability (i.e., are the lethal targets of beta-lactams), a priori knowledge of the borrelial PBPs will facilitate the production and purification of recombinant derivatives whose activities can be assessed further in vitro.

Ampicillin↗

Nisin as a model food preservative.

Nisin is a ribosomally synthesized peptide that has broad-spectrum antibacterial activity, including activity against many bacteria that are food-spoilage pathogens. Nisin is produced as a fermentation product of a food-grade bacterium, and the safety and efficacy of nisin as a food preservative have resulted in its widespread use throughout the world, including the U.S. Nisin is a member of the class of antimicrobial substances known as lantibiotics, so called because they contain the unusual amino acid lanthionine. Lantibiotics, in general, have considerable promise as food preservatives, although only nisin has been sufficiently well characterized to be used for this purpose. As the number of known natural lantibiotics has increased and their useful characteristics have been explored, it has become desirable to synthesize structural analogs of nisin and other lantibiotics that do not occur naturally. The fact that lantibiotics are gene-encoded peptides synthesized by transcription and translation allows structural variants to be generated by mutagenesis. This review focuses on the progress that has been made in the construction and biological expression of genetically engineered nisin structural analogs. For example, a host-vector pair has been engineered that permits the construction of mutants of the structural gene for subtilin, which is a naturally occurring structural analog of nisin. The vector is designed in such a way that the mutant gene can be substituted for the natural subtilin gene in the chromosome of Bacillus subtilis, which in turn directs the transcription, translation, posttranslational modifications, and secretion of the mature form of the structural analog. Several structural analogs have been constructed, and their properties have provided insight into some of the structure-function relationships in lantibiotics, as well as their mechanism of antimicrobial action. These advances are assessed together with potential problems in the future development of nisin analogs as valuable new food preservatives.

Amino Acid Sequence↗

Prognostic significance of the immunological variant in patients with multiple myeloma.

INTRODUCTION: The prognostic significance of the immunological variant and especially of the type of light chain secretion in multiple myeloma is controversially assessed in the literature. OBJECTIVE: To study the prognostic significance of the class and type of the monoclonal production in patients with multiple myeloma. PATIENTS AND METHODS: 109 patients with multiple myeloma were enrolled, in the study, diagnosed and treated at the University Clinic. The class and type of the paraprotein secretion was determined using immune electrophoresis of serum and/or urine by Grabar and Williams, Mancini radial immunodiffusion and Ouchterlony immunodiffusion for light chain typing. The median survival (MS) was calculated using the method of maximal logarithmic authenticity, where the survival in months = log t. RESULTS: The MS of all patients was 29 months. Patients with Bence Jones myelomas showed the lowest survival--12 months; followed by those with IgD myelomas--20 months. The patients with combined and non-combined light chain production (BJ+) lived 26 months, with BJ(-)--35 months (inauthentic difference). No difference was found between the survival of BJ(kappa)--30 months and BJ(lambda)--21 months. BJ(+) patients with myeloma nephropathy and nitrogen retention had a MS of 23 and 20 months, respectively, as compared with the BJ(+) patients without myeloma nephropathy and nitrogen retention: 47 and 40 months (p < 0.05). DISCUSSION AND CONCLUSION: The class of the paraprotein secretion only and the two types of light chain separately do not possess any prognostic significance. From a quantitative point of view the class and type of paraprotein production are independent staging criteria with certain predictive value. The prognostic significance of BJ-proteinuria is found making comparison between the groups in which its pathogenic effect is expressed.

Adult↗

Endotracheal tubes versus laryngeal mask airways in rabbit inhalation anesthesia: ease of use and waste gas emissions.

In this study, we compared two endotracheal tubes (cuffed [Murphy Eye type] and uncuffed [Cole type]) and a pediatric laryngeal mask airway (LMA) with respect to their ease of use in rabbits and their capacities to limit waste isoflurane emissions. Animals (New Zealand White, 3.3 to 5.0 kg, n = 8) were sedated with intramuscular ketamine (50 mg/kg) and xylazine (10 mg/kg). After 5 min, the larynx was numbed with cetocaine, an intubation device was positioned, and anesthesia was maintained with isoflurane (2%) in oxygen (1 liter/min). Real-time atmospheric isoflurane emissions were assessed at the rabbit's oral commissure and in the operator's breathing zone (45 cm from the rabbit's nose) by using a portable infrared spectrophotometer. The LMA was placed more easily than was either endotracheal tube, especially by novices. The cuffed tube was positioned more readily than was the uncuffed variant. All three devices emitted isoflurane. The concentrations measured at the oral commissure for the LMA (mean +/- standard error, 8.4 +/- 0.6 ppm) were modestly higher than those acquired for the cuffed (6.7 +/- 0.5 ppm) and uncuffed (6.3 +/- 0.4 ppm) endotracheal tubes; the difference between the LMA and uncuffed tube was significant (P = 0.012). Isoflurane was not detected in the operator's breathing zone. These data show that the uncuffed endotracheal tube (usually used to anesthetize birds and reptiles) and the pediatric LMA can be used in rabbits as readily as a cuffed tube. In addition, our findings indicate that tradeoffs will be required in selecting a delivery system for this species, as the easiest apparatus (the LMA) also emits the most isoflurane waste.

Anesthesia, Inhalation↗

Infection with Anaplasma phagocytophila in cervids from Slovenia: evidence of two genotypic lineages.

OBJECTIVE: Human granulocytic ehrlichiosis (HGE) was recently recognized as an emerging tick-borne infection in Europe. The disease is caused by Anaplasma (previously Ehrlichia) phagocytophila. The first confirmed acute human disease caused by A. phagocytophila was reported from Slovenia in 1998. The tick Ixodes ricinus was identified as the likely vector for this pathogen of humans and animals in Europe. In order to assess the possibility that roe and red deer in Slovenia serve as potential reservoir hosts for A. phagocytophila, materials from both species were examined. METHODS: Samples were obtained from 32 red deer (Cervus elaphus) and 56 roe deer (Capreolus capreolus). Polyvalent antibodies to the USG3 isolate of Anaplasma phagocytophila were detected by indirect immunofluorescence assay (IFA). DNA was extracted from spleen tissue. The 16S rRNA gene and a portion of the groESL heat shock operon were used for PCR detection and subsequent direct sequencing of amplified products. RESULTS: Serological and PCR results indicated that high proportions of roe and red deer were infected with A. phagocytophila. Infection was confirmed in 74% of the animals by IFA and in 86% of animals by PCR. While similar prevalences by PCR were seen in the two species (approximately 86%), the prevalence of antibodies was much higher in roe deer (94% vs. 35% in red deer). Sequence analysis of a 1256-bp fragment of the groESL operon revealed genetic diversity among collected samples. Identity of sequences ranging from 98% to 100%. None of the A. phagocytophila groESL and 16S rRNA gene sequences from red or roe deer were identical to the sequences previously obtained from human patients with ehrlichiosis from Slovenia or elsewhere in the world. All red deer sequences clustered with those obtained from humans, whereas all but two sequences from roe deer clustered separately. CONCLUSIONS: The results of our study indicate that a high percentage of red deer and roe deer in Slovenia are infected with A. phagocytophila. Analysis of groESL and 16S rRNA gene sequences revealed two distinct genetic lineages. Among deer, one variant was primarily associated with roe deer. Although none of the sequences from red deer was identical to those found in humans, they were more closely related.

Anaplasma phagocytophilum↗

The P9 peptide sidechain specificity of I-Ad.

The murine MHC class II variant I-Ad confers susceptibility to herpes simplex virus (HSV)-induced keratitis and relative protection against type 1 diabetes mellitus. The association to these autoimmune diseases appears to be largely determined by the peptide sidechain specificity of the P9 pocket, which we therefore have analyzed in detail. Assessment of T-cell responses and I-Ad binding capacity of position 446-substituted analogs of an IgG2a allotype b (IgG2a(b)) heavy chain peptide demonstrates that engagement of the P9 pocket is crucial for effective peptide presentation. Sidechain size rather than charge decides the capacity to engage the P9 pocket. Thus, small, uncharged sidechains are accepted, whereas acidic and aromatic amino acids as well as lysine and arginine are disfavored. The specificity of the P9 pocket of I-Ad (serine beta57) is distinct from that of the diabetes-associated I-Ag7 (aspartic acid beta57), supporting the contention that the polymorphism at residue beta57 influences diabetes susceptibility via P9-specific effects on the repertoires of self peptides presented to T cells. Furthermore, the data rationalize the susceptibility to HSV-induced keratitis conferred by the a and the protection conferred by the b allotypes of the IgG2a heavy chain. Keratitogenic T cells, which cross-react with the viral UL6 protein and a corneal antigen, are silenced in IgG2a(b) mice because of antigenic mimicry with gamma2a(b) 435-451. Our finding that the lysine P9 residue of the corresponding gamma2a(a) allopeptide precludes high-affinity binding to I-Ad indicates that the susceptibility of IgG2a(a) mice reflects inefficient thymic presentation of autologous IgG2a and thus failure to purge the T-cell repertoire of the pathogenic clones.

Animals↗

Benign lichenoid keratosis: a clinical and pathologic reappraisal of 1040 cases.

Benign lichenoid keratosis, otherwise known as lichen planus-like keratosis, is a common, cutaneous entity that is often confused with cutaneous malignancy. Few studies have examined the multiple clinical and pathologic guises of this entity, particularly within the context of clinical pathologic correlation or magnitude of this study. We examined the epidemiologic, clinical, and pathologic attributes of 1040 consecutive cases of benign lichenoid keratosis referred for pathologic examination at a busy laboratory over an entire year. Clinical parameters assessed included the age, anatomic location, gender, and multiplicity of the lesions. Pathologic attributes were assessed yielding discernment of five different subtypes that included a classic type, bullous type, atypical type with cytologically atypical lymphocytes, an early or interface type, and a late regressed or atrophic type. The results yielded an average age at presentation of 59.5 years with an age range of 36 to 87 years. The gender frequency was 76% female, 24% male. The trunk was the most common location (76%), followed by the extremities (33%) and head and neck (7%); 8% of patients presented with two lesions and less than 1% with three lesions prompting consideration of lichen planus. The classic, atypical, and bullous forms of the disease clinically presented with erythematous papule/plaque(s). The early or interface type showed erythematous to hyperpigmented brown macules and the regressed or atrophic type presented as violaceous papules or irregularly distributed macular pigmentation; 81% of the lesions showed the classic histology consisting of epidermal acanthosis with a band-like lichenoid lymphocytic infiltrate. Variable numbers of plasma cells, eosinophils, and neutrophils were identified as well as epidermal parakeratosis distinguishing these lesions from typical lichen planus. The bullous variant showed intraepidermal or subepidermal bullous cavities with a dense associated lymphocytic infiltrate and increased numbers of necrotic basilar layer keratinocytes. The atypical variant showed features of the classic type with scattered enlarged CD-3, CD-30 (+) lymphocytes possessing hyperchromatic, irregular nuclei. The early interface type showed single lymphocytes aligned along the dermoepidermal junction without epidermal acanthosis and adjacent lentigo. The regressed or atrophic variant showed epidermal atrophy with papillary dermal scarring, patchy lymphocytic infiltrates and melanin incontinence. The clinicopathologic spectrum of benign lichenoid keratosis is broad and encompasses several unrelated entities. An awareness of its expanded presentation is essential to avoid misdiagnosis and may serve as an important forerunner of pathogenic discernment.

Adult↗

APOL1 kidney disease: a critical narrative review of molecular mechanisms, clinical heterogeneity, and the emerging therapeutic landscape.

BACKGROUND: The G1 and G2 variants of the APOL1 gene represent significant genetic risk factors for APOL1 kidney disease and contribute substantially to the excess burden of renal disease observed in individuals of African ancestry. Importantly, both variants exhibit incomplete penetrance, with only approximately 15-20% of high-risk genotype carriers ultimately developing overt nephropathy. OBJECTIVE: To provide a critically appraised, clinically oriented narrative synthesis of APOL1 kidney disease that (i) assigns an explicit certainty rating to each major mechanistic and clinical claim, (ii) identifies where published estimates diverge, where associations remain contested, and where conclusions have been overstated in the secondary literature, and (iii) aligns terminology, testing guidance and therapeutic expectations with the conclusions of the 2025 KDIGO Controversies Conference and with clinical trial data available to August 2026. METHODS: This literature narrative review was performed using a literature search of PubMed and Scopus focusing on APOL1-related nephropathy. Mainly studies published from 2010 to 2026 were considered; however, some selected historical papers from 2005 to 2010 were used for better understanding of the underlying mechanisms and history. Used search terms were "APOL1," "APOL1 risk variants," "chronic kidney disease," AMPLITUDE trial, MZE829, HORIZON trial, "focal segmental glomerulosclerosis," "HIV-associated nephropathy," "podocyte injury," "inaxaplin," "VX-147," KDIGO 2025, and "antisense oligonucleotides." Trial status and topline results for agents in development were additionally verified against ClinicalTrials.gov registrations and sponsor disclosures. The literature search was last updated on 10 August 2026. The inclusion criteria of the study were peer-reviewed original articles, genome-wide association studies, randomised controlled trials, translational studies, mechanistic investigations, and high-quality review articles published in the English language. Exclusion criteria included conference abstracts without peer review, duplicate papers, non-English publications with unreliable translation, and case reports with no relevance to the underlying mechanisms. More attention was paid to studies focusing on molecular pathogenesis of APOL1 nephropathy, second-hit pathophysiology, genotypes/phenotypes, and new therapies (e.g. inhibitors such as Inaxaplin). The review method and design have been prepared according to SANRA (Scale for the Assessment of Narrative Review Articles) criteria. Among eligible articles, priority was given to studies with larger sample sizes, more recent publication dates, higher-impact peer-reviewed journals, and direct clinical or mechanistic relevance to APOL1-associated nephropathy; where multiple studies addressed the same question, the most methodologically rigorous and most recent source was preferentially cited. To move beyond description, each principal claim carried forward into this review was assigned a qualitative certainty rating (high, moderate, low or very low) on the basis of study design, consistency across independent cohorts, directness of the evidence to human disease, and precision of the estimate. These ratings, together with the study design that would be required to resolve each remaining uncertainty, are presented in Table&#xa0;5. This grading represents a structured judgement by the authors and is not a formal GRADE assessment. RESULTS: Pathogenic actions of APOL1 risk alleles depend on toxic gain-of-function activities that result from the disruption of ion channels. Mitochondrial dysfunction, endoplasmic reticulum stress, and inflammasome activation play roles as secondary downstream modulators of podocyte damage. The existence of incomplete penetrance and lack of symptoms in people with high-risk alleles highlights the need for secondary triggers, including environmental, infectious, and inflammatory factors, for disease onset and progression. High-risk APOL1 genotypes increase the likelihood of rapidly progressing kidney diseases like FSGS, which amplify susceptibility in HIVAN when accompanied by secondary causes like HIV infection. Management is mainly through renin-angiotensin antagonists, but recent treatments include antisense oligonucleotides, immunomodulators, and small molecule inhibitors like inaxaplin. Although promising, inaxaplin (VX-147) showed a ~47% reduction in urine protein/creatinine ratio (UPCR) in Phase 2a trial; however, these findings are based on a relatively small sample size, an open-label study design, and short-term follow-up, and therefore require confirmation in ongoing Phase 3 studies. As this is a narrative review rather than a primary study, no new patient-level data are reported. Across the studies synthesised, high-risk APOL1 genotypes were consistently associated with podocyte injury and with a faster decline in kidney function than low-risk genotypes; however, the magnitude of this association varied substantially with how cohorts were ascertained. The association is robust and reproducible for focal segmental glomerulosclerosis, HIV-associated nephropathy, and hypertension-attributed kidney failure, and remains inconsistent for diabetic kidney disease. Therapeutic development has accelerated, but the supporting clinical evidence remains early phase. Inaxaplin (VX-147) reduced the urine protein-to-creatinine ratio by approximately 47.6% at week 13 in a 16-participant, single-group, open-label Phase 2a study, and is now being evaluated in the randomised, double-blind, placebo-controlled Phase 2/3 AMPLITUDE trial (NCT05312879), whose pre-specified week 48 interim analysis is anticipated in early 2027. MZE829, an orally administered APOL1 inhibitor, produced a mean 35.6% reduction in the urine albumin-to-creatinine ratio at 12&#xa0;weeks in the Phase 2 HORIZON study; because HORIZON was a small, open-label, single-arm basket study (15 participants enrolled, 12 evaluable) whose primary endpoints were safety and tolerability, this reduction is neither placebo adjusted nor the result of a formal test of efficacy. To date, no APOL1-targeted agent has demonstrated benefit on a hard kidney endpoint. CONCLUSION: APOL1 is the clearest current example of a genetically defined, mechanism-targetable kidney disease, but its evidence base is uneven. The genetic association is firmly established; whereas much of the mechanistic literature derives from overexpression systems, several downstream pathways remain contested, and every APOL1-targeted therapy is so far supported only by short-term, surrogate-endpoint data. The principal unresolved issues are the determinants of incomplete penetrance, the absence of a validated progression biomarker and of any model reproducing the common slowly progressive phenotype, and the long-term efficacy and safety of APOL1-directed therapy. Genotype-guided risk stratification is therefore best regarded as clinically reasonable but not yet proven, and routine population-level screening is not currently supported.

AMPLITUDE trial↗

[Plasma chitotriosidase activity in Argentinian patients with Gaucher disease, various lysosomal diseases and other inherited metabolic disorders].

The striking and apparent specific elevation of the activity of chitotriosidase found in plasma from patients with Gaucher disease (GD) Type 1 (Mc Kusick 230800) is considered a new diagnostic hallmark of GD and should prove useful in assessing clinical manifestations and monitoring enzyme supplementation therapy. Further data have suggested that the increased levels of chitotriosidade activity in plasma from patients with unexplained diseases may be indicative of a lysosomal storage disorder. We present here an experience of the plasmatic chitotriosidase in an Argentinean population consisting of three groups: a) 25 healthy controls; b) subjects related with GD: 3 patients Type 1, 3 obligated heterocygotes and 1 patient with an atypical variant of GD; c) 42 patients with a precise nosologycal definition of inherited error of metabolism (IEM) and 5 patients presumably affected by a lysosomal pathology but without enzymatic confirmation. Methylumbellypheryl-tri-N-acetyl chitotrioside hydrolase activity was markedly increased: the mean activity being > 600 times and > 100 times the mean value in plasma of our healthy control (mean 17 nmol/min/ml, range 6-60.4 nmol/min/ml) in the plasma of the patients of Gaucher Type 1 disease and an atypical variant of GD, respectively. In the two more affected patients the elevated levels of chitotriosidase activity ran parallel to the severity of the disease and also as a response to the supplementation enzymatic therapy with a decrease of 50% of its activity at 10 months of therapy at a dose of 30 u/kg/month. Moreover, no increase of chitotriosidase activity was found in the 3 obligated heterozygotes of Gaucher Type 1 and Type 2 disease or in any other of the IEM. Chitotriosidase activity was absent in plasma of 2 control subjects and in 4 patients with exact diagnosis of an IEM. The physiological role of the human chitinase still has to be established and warrants further investigation and the natural consequence of the high frequency of a genetic deficiency in man. Meanwhile, reports on the purification and characterization of human chitotriosidase with chitinolytic activity toward chitin-containing pathogens and on the cloning of cDNA encoding chitinase will be crucial to obtain a better insight into this new chapter of medical genetics.

Adult↗

Variant Creutzfeldt-Jakob disease: risk of transmission by blood transfusion and blood therapies.

In the last decade, a new variant of the human prion disease Creutzfeldt-Jakob disease (now known as variant CJD or vCJD) was identified and causally linked to dietary exposure to bovine spongiform encephalopathy (BSE) during the 1980s and early 1990s. Preliminary studies in animal models suggest that prions can be transmitted by blood. Based on two recent reports of iatrogenic vCJD transmission by blood transfusion in humans, a Department of Health-sponsored risk assessment warned that recipients of plasma therapies are now at risk of contracting vCJD from potentially infected donors. It is believed that all the population may be susceptible to vCJD infection, although clinical cases have so far occurred only in methionine homozygotes at codon 129 in the human prion protein gene. A non-invasive blood-based diagnostic assay is urgently needed. Because the incubation period may be upwards of 40 years and there is no reliable screening test, it is currently unknown how many people may be in an asymptomatic phase of vCJD infection in the UK. However, there remains a distinct possibility that some infected patients may never develop clinical symptoms but will remain asymptomatic carriers who can potentially transmit the disease to other individuals. Therefore, screening of infectious individuals will be a critical component for individuals who rely on blood transfusions and/or blood therapies. In the absence of screening tests or effective therapies to treat this disease, a formidable worldwide public health challenge lies ahead to prevent new infections, accurately assess infection rates and treat infected patients.

Animals↗

Risks of non-breast, non-ovarian cancers for BRCA1 and BRCA2&#xa0;pathogenic variant carriers: a prospective cohort study.

BACKGROUND: The non-breast non-ovarian cancers associated with BRCA1 and BRCA2 pathogenic variants (PVs) are controversial. We aimed to examine this using a prospective cohort&#xa0;design. METHODS: This study included 1260 BRCA1 and 1058 BRCA2 PV carriers (91% were females) from two consortia: the Breast Cancer Family Registry (BCFR) and the Kathleen Cuningham Foundation Consortium for Research into Familial Breast Cancer Follow-Up Study (kConFab-FUS). The carriers were free of cancer other than breast or ovarian cancer&#xa0;at baseline and had a median baseline age of 45.5&#xa0;years. For 16 types of non-breast, non-ovarian cancers, standardized incidence ratios (SIRs) relative to population incidence, the probabilities of relative risk effect size&#x2009;>&#x2009;2 (i.e., moderate risk) and cumulative risks to age 80&#xa0;years were estimated. RESULTS: During a median follow-up time of 11.4&#xa0;years, 161 non-breast, non-ovarian cancers were observed. For BRCA1 PV carriers, little evidence of increased risk was observed. The prostate, pancreatic, and all non-pancreatic cancer SIRs were 1.7 (95% CI 0.7-4.2), 1.1 (95% CI 0.3-4.6) and 0.85 (95% CI 0.68-1.06), respectively; the probabilities of relative risk&#x2009;>&#x2009;2 were 0 and 67% for prostate and pancreatic cancers, respectively. For BRCA2 PV carriers, increased risks of pancreatic (SIR&#x2009;=&#x2009;6.6, 95% CI 3.8-11.6), prostate (SIR&#x2009;=&#x2009;3.6, 95% CI 1.9-6.8) and stomach (SIR&#x2009;=&#x2009;3.1, 95% CI 1.01-9.8) cancer were observed, with a cumulative risk to age 80&#xa0;years of 8.3, 82.0, and 1.6%, respectively. For all the other non-breast, non-ovarian cancers combined, the SIR was 0.85 (95% CI 0.66-1.10). CONCLUSIONS: Apart from pancreatic, prostate, and possibly stomach cancers for BRCA2 PV carriers, and possibly pancreatic cancer for BRCA1 PV carriers, there is no evidence that BRCA1 and BRCA2 PV carriers have substantially increased risks of other non-breast, non-ovarian cancers. Our prospective risk estimates are informative for cancer risk assessment for people with BRCA1 and BRCA2 PVs.

Humans↗