Two-Dimensional Spin-Crossover Molecular Solid Solutions with Tunable Transition Temperatures across 90 K.

Spin-crossover (SCO) materials exhibit remarkable potential as bistable switches in molecular devices. However, the spin transition temperatures (Tc) of known compounds are unable to cover the entire ambient temperature spectrum, largely limiting their practical utility. This study reports an exemplary two-dimensional SCO solid solution system, [FeIII(H0.5LCl)2-2x(H0.5LF)2x]·H2O (H0.5LX = 5-X-2-hydroxybenzylidene-hydrazinecarbothioamide, X = F or Cl, x = 0 to 1), in which the adjacent layers are adhered via hydrogen bonding. Notably, the Tc of this system can be fine-tuned across 90 K (227-316 K) in a linear manner by modulating the fraction x of the LF ligand. Elevating x results in strengthened hydrogen bonding between adjacent layers, which leads to enhanced intermolecular interactions between adjacent SCO molecules. Single-crystal diffraction analysis and periodic density functional theory calculations revealed that such a special kind of alteration in interlayer interactions strengthens the FeIIIN2O2S2 ligand field and corresponding SCO energy barrier, consequently resulting in increased Tc. This work provides a new pathway for tuning the Tc of SCO materials through delicate manipulation of molecular interactions, which could expand the application of bistable molecular solids to a much wider temperature regime.

USP3: Key deubiquitylation enzyme in human diseases.

Ubiquitination and deubiquitylation are pivotal posttranslational modifications essential for regulating cellular protein homeostasis and are implicated in the development of human diseases. Ubiquitin-specific protease 3 (USP3), a member of the ubiquitin-specific protease family, serves as a key deubiquitylation enzyme, playing a critical role in diverse cellular processes including the DNA damage response, cell cycle regulation, carcinogenesis, tumor cell proliferation, migration, and invasion. Despite notable research efforts, our current understanding of the intricate and context-dependent regulatory networks governing USP3 remains incomplete. This review aims to comprehensively synthesize existing published works on USP3, elucidating its multifaceted roles, functions, and regulatory mechanisms, while offering insights for future investigations. By delving into the complexities of USP3, this review strives to provide a foundation for a more nuanced understanding of its specific roles in various cellular processes. Furthermore, the exploration of USP3's regulatory networks may uncover novel therapeutic strategies targeting this enzyme in diverse human diseases, thereby holding promising clinical implications. Overall, an in-depth comprehension of USP3's functions and regulatory pathways is crucial for advancing our knowledge and developing targeted therapeutic approaches for human diseases.

AcHZP45 is a repressor of chlorophyll biosynthesis and activator of chlorophyll degradation in kiwifruit.

The degradation of chlorophyll during fruit development is essential to reveal a more 'ripe' color that signals readiness to wild dispersers of seeds and the human consumer. Here, comparative biochemical analysis of developing fruit of Actinidia deliciosa cv. Xuxiang ('XX', green-fleshed) and Actinidia chinensis cv. Jinshi No.1 ('JS', yellow-fleshed) indicated that variation in chlorophyll content is the major contributor to differences in flesh color. Four differentially expressed candidate genes were identified: the down-regulated genes AcCRD1 and AcPOR1 involved in chlorophyll biosynthesis, and the up-regulated genes AcSGR1 and AcSGR2 driving chlorophyll degradation. Prochlorophyllide and chlorophyllide, the metabolites produced by AcCRD1 and AcPOR1, progressively reduced in 'JS', but not in 'XX', indicating that chlorophyll biosynthesis was less active in yellow-fleshed fruit. AcSGR1 and AcSGR2 were verified to be involved in chlorophyll degradation, using both transient expression in tobacco and stable overexpression in kiwifruit. Furthermore, a homeobox-leucine zipper (HD-Zip II), AcHZP45, showed significantly increased expression during 'JS' fruit ripening, which led to both repressed expression of AcCRD1 and AcPOR1 and activated expression of AcSGR1 and AcSGR2. Collectively, the present study indicated that different dynamics of chlorophyll biosynthesis and degradation coordinate the changes in chlorophyll content in kiwifruit flesh, which are orchestrated by the key transcription factor AcHZP45.

Strategic exploration of whole grain cereals in modulating the glycaemic response.

In the current context, diabetes presents itself as a widespread and complex global health issue. This study explores the significant influence of food microstructure and food matrix components interaction (protein, lipid, polyphenols, etc.) on the starch digestibility and the glycaemic response of post-prandial glycemia, focusing on the potential effectiveness of incorporating bioactive components from whole grain cereals into dietary strategies for the management and potential prevention of diabetes. This study aims to integrate the regulation of postprandial glycaemic homeostasis, including the complexities of starch digestion, the significant potential of bioactive whole grain components and the impact of food processing, to develop a comprehensive framework that combines these elements into a strategic approach to diabetes nutrition. The convergence of these nutritional strategies is analyzed in the context of various prevalent dietary patterns, with the objective of creating an accessible approach to mitigate and prevent diabetes. The objective remains to coalesce these nutritional paradigms into a coherent strategy that not only addresses the current public health crisis but also threads a preventative approach to mitigate future prevalence and impact.

TEAD1, MYO7A and NDUFC2 are novel functional genes associated with glucose metabolism in BXD recombinant inbred population.

AIM: The liver is an important metabolic organ that governs glucolipid metabolism, and its dysfunction may cause non-alcoholic fatty liver disease, type 2 diabetes mellitus, dyslipidaemia, etc. We aimed to systematic investigate the key factors related to hepatic glucose metabolism, which may be beneficial for understanding the underlying pathogenic mechanisms for obesity and diabetes mellitus. MATERIALS AND METHODS: Oral glucose tolerance test (OGTT) phenotypes and liver transcriptomes of BXD mice under chow and high-fat diet conditions were collected from GeneNetwork. QTL mapping was conducted to pinpoint genomic regions associated with glucose homeostasis. Candidate genes were further nominated using a multi-criteria approach and validated to confirm their functional relevance in vitro. RESULTS: Our results demonstrated that plasma glucose levels in OGTT were significantly affected by both diet and genetic background, with six genetic regulating loci were mapped on chromosomes 1, 4, and 7. Moreover, TEAD1, MYO7A and NDUFC2 were identified as the candidate genes. Functionally, siRNA-mediated TEAD1, MYO7A and NDUFC2 knockdown significantly decreased the glucose uptake and inhibited the transcription of genes related to insulin and glucose metabolism pathways. CONCLUSIONS: Our study contributes novel insights to the understanding of hepatic glucose metabolism, demonstrating the impact of TEAD1, MYO7A and NDUFC2 on mitochondrial function in the liver and their regulatory role in maintaining in glucose homeostasis.

Copper-Catalyzed Domino-Double Annulation of o-Aminobenzamides with 2-Iodoisothiocyanates for the Synthesis of 12H-Benzo[4,5]thiazolo[2,3-b]quinazolin-12-ones.

A facile and efficient copper-catalyzed domino-double annulation strategy was developed from easily accessible o-aminobenzamides and 2-iodoisothiocyanates, which affords a direct pathway for the synthesis of tetracyclic fused 12H-benzo[4,5]thiazolo[2,3-b]quinazolin-12-ones in moderate to good yields without the addition of ligands, bases, and external oxidants. The reaction involves a C-N bond cleavage and the formation of a C-N/C-S bond in one step with the advantages of using an inexpensive copper catalyst and easy operation. Mechanistic studies suggest that this transformation proceeds via intermolecular condensation of o-aminobenzamides with 2-iodoisothiocyanates, followed by an intramolecular Ullmann-type cross-coupling cyclization reaction.

A nomogram prediction of implant apical non-coverage on bone-added transcrestal sinus floor elevation: A retrospective cohort study.

OBJECTIVES: To identify the risk indicators and develop and validate a nomogram prediction model of implant apical non-coverage by comprehensively analyzing clinical and radiographic factors in bone-added transcrestal sinus floor elevation (TSFE). MATERIAL AND METHODS: A total of 260 implants in 195 patients receiving bone-added TSFE were included in the study. The population was divided into a development (180 implants) and a validation (80 implants) cohort. According to 6 months post-surgery radiographic images, implants were categorized as "apical non-coverage" or "apical covered." The association of risk factors including clinical and radiographic parameters with implant apical non-coverage was assessed using regression analyses. A nomogram prediction model was developed, and its validation and discriminatory ability were analyzed. RESULTS: The nomogram predicting bone-added TSFE's simultaneously placed implant's apex non-coverage after 6 months. This study revealed that sinus angle, endo-sinus bone gain, implant protrusion length, graft contact walls, and distal angle were predictors of implant apical non-coverage. The generated nomogram showed a strong predictive capability (area under the curve [AUC] = 0.845), confirmed by internal validation using 10-fold cross-validation (Median AUC of 0.870) and temporal validation (AUC = 0.854). The calibration curve and decision curve analysis demonstrated good performance and high net benefit of the nomogram, respectively. CONCLUSIONS: The clinical implementation of the present nomogram is suitable for predicting the apex non-coverage of implants placed simultaneously with bone-added TSFE after 6 months.

A further specification of the effects of font emphasis on reading comprehension: Evidence from event-related potentials and neural oscillations.

The attention hypothesis, which assumes that font emphasis captures readers' attention, is usually used to explain the mechanism by which such emphasis operates. This study further delineates the attention hypothesis by investigating the ways in which font emphasis captures attention and its effects on the integration of emphasized information into the previous context. We computed event-related potentials and frequency band-specific electroencephalographic power changes occurring while participants read sentences containing critical words that were either emphasized (i.e., displayed in a color different from the other words in the sentence) or not (i.e., shown in the same color as the rest of the sentence) and semantically congruent with prior words or not. The results showed that the emphasized words (as compared to control words) elicited a reduced N1 and increased P2, indicating that font emphasis reduced familiarity-based visuo-orthographic processing and instead increased controlled attentional processing. We also observed greater P300 and power decreases in the alpha and beta frequency range in response to critical words in the emphasized condition, suggesting that font emphasis enhances focal attention to promote a fuller integration of information into the sentence context. Furthermore, relative to the control condition, the emphasized condition induced delta and theta power increases for the incongruent words. These results suggest that font emphasis increases the efficiency of glyph processing, which facilitates lexical access.

Nursing Care of Postoperative Supplementary Surgery for Patients with Positive or High-level CIN after Conization Using CINIII Cold Knife.

Objective: This study aimed to evaluate the effectiveness of personalized nursing care in postoperative supplementary surgery for cervical intraepithelial neoplasia grade III (CIN III) patients with positive or high-grade CIN margins after cold knife conization at the Fourth Hospital of Hebei Medical University in Shijiazhuang, China. The primary objective was to assess the impact of personalized nursing intervention on postoperative outcomes, including psychological well-being, self-care ability, quality of life, and postoperative complications. Methods: A total of 106 patients undergoing additional surgery after CIN III cold knife conization at the Fourth Hospital of Hebei Medical University from January 2020 to April 2023 were randomly allocated into an observation group (n=53) and a control group (n=53) using a random number table method. Detailed information on the randomization process, including stratification factors and blinding procedures, is provided. The observation group received personalized nursing intervention, while the control group received routine nursing. The retreatment methods, including repeat cold knife conization, total hysterectomy, and radical cervical cancer surgery, were analyzed using appropriate statistical methods. Statistical software was employed for data analysis. Results: Pathological results post-cold knife conization revealed positive margins in 76 cases and pathological upgrades in 30 cases. The consistency rate between post-cold knife conization and post-retreatment pathological results was 75.47%. After the intervention, the observation group exhibited significantly lower SAS and SDS scores and significantly higher ESCA and WHO QOL-100 scores compared to the control group (P < .05), indicating improved psychological well-being and quality of life. The total incidence of postoperative complications in the observation group was 5.66%, significantly lower than in the control group (P < .05). Conclusion: Personalized nursing care in postoperative supplementary surgery for CIN III patients with positive or high-grade CIN margins at the Fourth Hospital of Hebei Medical University improves psychological well-being, self-care ability, quality of life, and reduces postoperative complications. The findings underscore the importance of tailored nursing interventions in enhancing patient outcomes. The inclusion of detailed patient demographics and methodological transparency enhances the generalizability and reliability of the study findings beyond the study setting.

lncRNA MAGI2-AS3 suppresses castration-resistant prostate cancer proliferation and migration via the miR-106a-5p/RAB31 axis.

Prostate cancer (PCa) is a common malignant cancer in elderly males in Western countries. Whole-genome sequencing confirmed that long non-coding RNAs (lncRNAs) are frequently altered in castration-resistant prostate cancer (CRPC) and promote drug resistance to cancer therapy. Therefore, elucidating the prospective role of lncRNAs in PCa oncogenesis and progression is of remarkable clinical significance. In this study, gene expression in prostate tissues was determined using RNA-sequencing datasets, and the gene diagnostic and prognostic values of CRPC were analyzed using bioinformatics. Further, the expression levels and clinical significance of MAGI2 Antisense RNA 3 (MAGI2-AS3) in PCa clinical specimens were evaluated. The tumor-suppressive activity of MAGI2-AS3 was functionally explored in PCa cell lines and animal xenograft models. MAGI2-AS3 was found to be aberrantly decreased in CRPC and was negatively correlated with Gleason score and lymph node status. Notably, low MAGI2-AS3 expression positively correlated with poorer survival in patients with PCa. The overexpression of MAGI2-AS3 significantly inhibited the proliferation and migration of PCa in vitro and in vivo. Mechanistically, MAGI2-AS3 could play a tumor suppressor function in CRPC through a novel miR-106a-5p/RAB31 regulatory network and could be a target for future cancer therapy.

The effects of environmental factors on the synthesis of water-soluble Monascus red pigments via submerged fermentation: a review.

Monascus pigments (MPs) have been used as natural food pigments for many years. There is a high demand for Monascus red pigments (MRPs) to enhance color and for antibacterial and cancer prevention therapies in food and medicine. Most MRPs are not water soluble, and the yield of water-soluble MRPs is naturally low. On the other hand, water-soluble MRP is more cost effective for application in industrial mass production. Therefore, it is important to improve the yield of water-soluble MRPs. Environmental factors have a significant influence on the synthesis of water-soluble MRPs, which is crucial for the development of industrial production of water-soluble MRPs. This review introduces the biosynthetic pathways of water-soluble MRPs and summarizes the effects of environmental factors on the yield of water-soluble MRPs. Acetyl coenzyme A (acetyl-CoA) is a precursor for MPs synthesis. Carbon and nitrogen sources and the carbon/nitrogen ratio can impact MP production by regulating the metabolic pathway of acetyl-CoA. Optimization of fermentation conditions to change the morphology of Monascus can stimulate the synthesis of MPs. The appropriate choice of nitrogen sources and pH values can promote the synthesis of MRPs from MPs. Additives such as metal ions and non-ionic surfactants can affect the fluidity of Monascus cell membrane and promote the transformation of MRPs into water-soluble MRPs. This review will lay the foundation for the industrial production of water-soluble MRPs. © 2024 Society of Chemical Industry.

Effect of GABA combined with ultrasound stress germination treatment on phenolic content and antioxidant activity of highland barley.

BACKGROUND: This study investigated the effects of γ-aminobutyric acid (GABA) combined with ultrasonic stress germination (AUG) treatment on the phenolic content and antioxidant activity of highland barley (HB). Key variables, including germination times (ranging from 0 to 96 h), ultrasonic power (200-500 W), and GABA concentration (5-20 mmol/L), were optimized using response surface methodology (RSM) to enhance the enrichment of phenolic compounds. Furthermore, the study assessed the content, composition, and antioxidant activities of phenolic compounds in HB under various treatment conditions such as germination alone (G), ultrasonic stress germination (UG), and AUG treatment. RESULTS: The study identified optimal conditions for the phenolic enrichment of HB, which included a germination time of 60 h, an ultrasound power of 300 W, and a GABA concentration of 15 mmol L-1. Under these conditions, the total phenolic content (TPC) in HB was measured at 7.73 milligrams of gallic acid equivalents per gram dry weight (mg GAE/g DW), representing a 34.96% enhancement compared to untreated HB. Notably, all treatment modalities - G, UG, and AUG - significantly increased the phenolic content and antioxidant activity in HB, with the AUG treatment proving to be the most effective. CONCLUSION: These obtained results suggest that AUG treatment is a promising processing method for enriching phenolic compounds and improving antioxidant activity in HB. Subsequently, the AUG-treated HB can be used to develop phenolic-rich germinated functional foods to further broaden the application of HB. © 2024 Society of Chemical Industry.

Association between phthalates and sleep problems in the U.S. adult females from NHANES 2011-2014.

There is little research on the relationship between phthalates exposure and sleep problems in adult females, with existing studies only assessing the association between exposure to individual phthalates with sleep problems. We aimed to analyse the relationship between phthalates and sleep problems in 1366 US females aged 20 years and older from the 2011-2014 National Health and Nutrition Examination Survey (NHANES) by age stratification. Multivariate logistic regression showed that the fourth quartile of MECPP increased the risk of sleep problems in females aged 20-39 compared with the reference quartile (OR: 1.87, 95% CI: 1.14, 3.08). The WQS index was significantly associated with the sleep problems in females aged 20-39. In the BKMR, a positive overall trend between the mixture and sleep problems in females aged 20-39. In this study, we concluded that phthalates might increase the risk of sleep problems in females aged 20-39.

Revealing Intramolecular Isotope Effects with Chemical-Bond Precision.

Isotope substitution of a molecule not only changes its vibrational frequencies but also changes its vibrational distributions in real-space. Quantitatively measuring the isotope effects inside a polyatomic molecule requires both energy and spatial resolutions at the single-bond level, which has been a long-lasting challenge in macroscopic techniques. By achieving ångström resolution in tip-enhanced Raman spectroscopy (TERS), we record the corresponding local vibrational modes of pentacene and its fully deuterated form, enabling us to identify and measure the isotope effect of each vibrational mode. The measured frequency ratio νH/νD varies from 1.02 to 1.33 in different vibrational modes, indicating different isotopic contributions of H/D atoms, which can be distinguished from TERS maps in real-space and well described by the potential energy distribution simulations. Our study demonstrates that TERS can serve as a non-destructive and highly sensitive methodology for isotope detection and recognition with chemical-bond precision.

Dynamic Metal-Ligand Coordination Boosts CO2 Electroreduction.

The interfacial structure of heterogeneous catalysts determines the reaction rate by adjusting the adsorption behavior of reaction intermediates. Unfortunately, the catalytic performance of conventionally static active sites has always been limited by the adsorbate linear scaling relationship. Herein, we develop a triazole-modified Ag crystal (Ag crystal-triazole) with dynamic and reversible interfacial structures to break such a relationship for boosting the catalytic activity of CO2 electroreduction into CO. On the basis of surface science measurements and theoretical calculations, we demonstrated the dynamic transformation between adsorbed triazole and adsorbed triazolyl on the Ag(111) facet induced by metal-ligand conjugation. During CO2 electroreduction, Ag crystal-triazole with the dynamically reversible transformation of ligands exhibited a faradic efficiency for CO of 98% with a partial current density for CO as high as -802.5 mA cm-2. The dynamic metal-ligand coordination not only reduced the activation barriers of CO2 protonation but also switched the rate-determining step from CO2 protonation to the breakage of C-OH in the adsorbed COOH intermediate. This work provided an atomic-level insight into the interfacial engineering of the heterogeneous catalysts toward highly efficient CO2 electroreduction.

Effect and mechanism of hydrogen-rich bath on mice with imiquimod-induced psoriasis.

The purpose of this study was to investigate whether hydrogen-rich bath has therapeutic effect on psoriasis and its molecular mechanism. Mice with imiquimod-induced psoriasis were established and divided into groups. The mice were respectively treated with hydrogen-rich water bath and distilled water bath. The changes of skin lesions and PSI scores of mice were compared after their treatments. HE staining was used to observe the pathological feature. The changes of inflammatory indexes and immune factors were analysed by ELISA and immunohistochemical staining. Malondialdehyde (MDA) content was measured by the thiobarbituric assay (TBA) method. By naked eye, the severity of skin lesions in hydrogen-rich water bath group was lower than that in distilled water bath group, and the psoriasis severity index (PSI) was lower (p < 0.01). The results of HE staining showed that the mice with distilled water bath had more abnormal keratosis, thickening of the spinous layer and prolongation of the dermal process, and more Munro abscess than the mice with hydrogen-rich water bath. During the course of disease, the overall levels and peaks of IL-17, IL-23, TNF-α, CD3+ and MDA in mice with hydrogen-rich bath were lower than those in mice with distilled water bath (p < 0.05). In the skin, the mice treated with the hydrogen-rich water bath also had lower peak of proliferating cell nuclear antigen (PCNA) levels. It is concluded that hydrogen-rich water bath can inhibit psoriasis inflammation and oxidative stress, relieve psoriasis skin lesions and accelerate the end of abnormal skin proliferation state, which shows a therapeutic and improving effect on psoriasis.

Ubiquitous Superconducting Diode Effect in Superconductor Thin Films.

The macroscopic coherence in superconductors supports dissipationless supercurrents that could play a central role in emerging quantum technologies. Accomplishing unequal supercurrents in the forward and backward directions would enable unprecedented functionalities. This nonreciprocity of critical supercurrents is called the superconducting (SC) diode effect. We demonstrate the strong SC diode effect in conventional SC thin films, such as niobium and vanadium, employing external magnetic fields as small as 1 Oe. Interfacing the SC layer with a ferromagnetic semiconductor EuS, we further accomplish the nonvolatile SC diode effect reaching a giant efficiency of 65%. By careful control experiments and theoretical modeling, we demonstrate that the critical supercurrent nonreciprocity in SC thin films could be easily accomplished with asymmetrical vortex edge and surface barriers and the universal Meissner screening current governing the critical currents. Our engineering of the SC diode effect in simple systems opens the door for novel technologies while revealing the ubiquity of the Meissner screening effect induced SC diode effect in superconducting films, and it should be eliminated with great care in the search for exotic superconducting states harboring finite-momentum Cooper pairing.

Copper-Promoted Aerobic Oxidative [3+2] Cycloaddition Reactions of N,N-Disubstituted Hydrazines with Alkynoates: Access to Substituted Pyrazoles.

A copper-promoted aerobic oxidative [3+2] cycloaddition reaction for the synthesis of various substituted pyrazoles from N,N-disubstituted hydrazines with alkynoates in the presence of bases is developed. This work involves a direct C(sp3)-H functionalization and the formation of new C-C/C-N bonds. In this strategy, inexpensive and easily available Cu2O serves as the promoter and air acts as the green oxidant. The reaction exhibits the advantages of high atom and step economy, high regioselectivity, and easy operation.

DBU-Promoted Deaminative Thiolation of 1H-Benzo[d]imidazol-2-amines and Benzo[d]oxazol-2-amines.

A facile and efficient catalyst-/metal-/oxidant-free DBU-promoted deaminative thiolation reaction of 1H-benzo[d]imidazol-2-amines and benzo[d]oxazol-2-amines has been developed at room temperature conditions in a one-pot protocol. This practical three-component strategy represents a novel and environmentally friendly reaction pathway toward the straightforward synthesis of various 2-thio-1H-benzo[d]imidazoles and 2-thiobenzo[d]oxazoles using carbon disulfide as a sulfur source through C-N bond cleavage and C-S bond formation process. Different types of 1H-benzo[d]imidazol-2-amines, benzo[d]oxazol-2-amines, and organic bromides are suitable substrates. The gram-scale and late-stage modification experiments provide the potential applications based on this methodology in the field of organic and medicinal chemistry.

Large deletion of Wdr19 in developing renal tubules disrupts primary ciliogenesis, leading to polycystic kidney disease in mice.

WD repeat domain 19 (Wdr19) is a major component of the intraflagellar transport (IFT) machinery, which is involved in the function of primary cilia. However, the effects of Wdr19 on primary cilia formation, cystogenesis, and polycystic kidney disease (PKD) progression remain unclear. To study these effects, we generated three lines of kidney-specific conditional knockout mice: Wdr19-knockout (Wdr19-KO, Wdr19f/- ::Cdh16-CreTg/0 ), Pkd1-knockout (Pkd1-KO, Pkd1f/- ::Cdh16-CreTg/0 ), and Wdr19/Pkd1-double knockout (Wdr19&Pkd1-dKO, Wdr19f/- ;Pkd1f/- ::Cdh16-CreTg/0 ) mice. Ultrastructural analysis using transmission electron microscopy (TEM) indicated that the primary cilia were almost absent at postnatal day 10 in Wdr19-KO mice compared with Pkd1-KO and wild-type (WT) mice. However, the primary cilia appeared structurally normal even if malfunctional in Pkd1-deficient cysts. The Pkd1-KO mice had the most severe PKD progression, including the shortest lifespan (14 days) and the largest renal cysts, among the three knockout lines. Thus, the molecular mechanism of renal cystogenesis in Wdr19-KO mice (primary cilia abrogation) was different from that in Pkd1-KO mice (primary cilia malfunction). In summary, Wdr19 deficiency leads to primary cilia abrogation and renal cyst formation. Wdr19 is primarily proposed to participate in retrograde IFT and to be crucial for the construction of primary cilia, which are critical organelles for tubulogenesis in the developing kidneys. © 2022 The Pathological Society of Great Britain and Ireland.