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Background: We conducted a cross-sectional study to examine two-leg- and one-leg-type balance characteristics in athletes and explore factors related to their balance ability., Methods: A total of 213 participants, including athletes from various sports (gymnastics, boat racing, swimming, soccer, judo, and baseball) and non-athletes, were included (142 men, 71 women, average age 21.5 ± 2.1 years). Balance ability was classified into two-leg and one-leg types using the modified index of postural stability (mIPS) in a two-leg stance and the one-legged stance duration with eyes closed (OLS). Body composition, upper and lower limb strength, and lower limb sensation were also measured. To examine the balance characteristics of each sport, the mIPS and OLS were used as dependent variables in a multiple regression model with age, height, weight, and sex as independent variables., Results: The results showed a significantly higher mIPS in gymnastics (estimate: 0.22) and boat racing (0.14), and it was lower in swimming (-0.25). The OLS was significantly higher in soccer (16.98), judo (16.23), gymnastics (9.77), and baseball (9.12) and significantly lower in swimming (7.93). Additionally, the mIPS was independently associated with knee extension strength (0.12), sensory motor variables (-0.004), and height (-0.01). The OLS was associated with skeletal muscle mass (1.85) and height (-1.42)., Conclusions: In summary, gymnasts showed superior two-leg and one-leg balance; boat racers excelled in two-leg balance; swimmers showed inferior two-leg but better one-leg balance; and soccer, judo, and baseball athletes demonstrated superior one-leg balance. Additionally, the mIPS was associated with knee extensor strength, plantar pressure sensation, and height, whereas the OLS was associated with skeletal muscle mass and height.

Postherpetic neuralgia (PHN) is characterized by persistent pain following the resolution of a herpes zoster rash. PHN is often resistant to treatment and can significantly reduce the patient's quality of life. Effective symptom relief is crucial and various treatments, including pharmacotherapy, have been attempted. Given that symptoms can persist for a prolonged period, they can substantially affect the physical and mental well-being of the patients. A 73-year-old man developed herpes zoster while undergoing treatment for a head angiosarcoma. Despite the resolution of the rash, the pain persisted, leading to the diagnosis of PHN. Treatment was initiated with a range of medications, including mecobalamin, pregabalin, and a combination of tramadol and acetaminophen, along with general pain relievers standardized as WHO Step 1 medications, which include acetaminophen and non-steroidal anti-inflammatory drugs (NSAIDs). However, achieving adequate pain control is challenging and results in frequent hospitalizations. Due to the patient's depression and the concurrent use of a selective serotonin reuptake inhibitor, duloxetine hydrochloride could not be prescribed. Instead, opioid therapy with continuous fentanyl citrate infusion was initiated. Eventually, the treatment was switched to oxycodone hydrochloride, which successfully stabilized the patient's symptoms. The use of conservative measures such as hot compresses also contributes to symptom relief. Alleviating pain symptoms using a combination of pharmacotherapeutic and non-pharmacological treatments is extremely important., Competing Interests: Human subjects: Consent was obtained or waived by all participants in this study. Conflicts of interest: In compliance with the ICMJE uniform disclosure form, all authors declare the following: Payment/services info: All authors have declared that no financial support was received from any organization for the submitted work. Financial relationships: All authors have declared that they have no financial relationships at present or within the previous three years with any organizations that might have an interest in the submitted work. Other relationships: All authors have declared that there are no other relationships or activities that could appear to have influenced the submitted work., (Copyright © 2024, Tsubaki et al.)

Vitamin B12 deficiency can cause a variety of diseases. The most common disease is macrocytic anemia, but it has also been found to be a cause of psychiatric disorders. The causes of deficiency are varied, and diagnosis is often difficult. Here, we report a patient who developed mental disorders due to vitamin B12 deficiency after total gastrectomy. A 37-year-old female, eight years after total gastrectomy, was withdrawn at her workplace, talking and acting abnormally. The family had seen unusual behavior for three days. The patient had no particular history of mental illness. The possibility of herpes encephalitis was suspected, and the patient was referred to our hospital, but there were no specific findings in the head on imaging examination. Blood tests showed no macrocytic anemia. Spinal fluid cytology and electroencephalography showed no specific findings, and herpes DNA was negative. Metabolic factors such as vitamin deficiency were considered, and intravenous vitamin replacement therapy was initiated. The psychiatric symptoms improved rapidly after vitamin B12 supplementation was started. On the fifth day of her hospitalization, it was discovered that her vitamin B12 level at the time of admission was extremely low. Typically, vitamin B12 deficiency is associated with macrocytic anemia, but in this patient, serum iron was also decreased, indicating a mixed anemia, making the diagnosis difficult. The patient had undergone a total gastrectomy for gastric cancer eight years ago, and the psychiatric symptoms were thought to be due to impaired vitamin B12 absorption caused by intrinsic factor deficiency. Since then, oral replacement therapy and intramuscular injection have been continued without recurrence of symptoms. Disturbances of consciousness may have many causes, but when there is a history of gastrectomy, we should also consider vitamin B12 deficiency when examining patients., Competing Interests: Human subjects: Consent was obtained or waived by all participants in this study. Conflicts of interest: In compliance with the ICMJE uniform disclosure form, all authors declare the following: Payment/services info: All authors have declared that no financial support was received from any organization for the submitted work. Financial relationships: All authors have declared that they have no financial relationships at present or within the previous three years with any organizations that might have an interest in the submitted work. Other relationships: All authors have declared that there are no other relationships or activities that could appear to have influenced the submitted work., (Copyright © 2024, Tsubaki et al.)

Signal regulatory protein alpha (SIRPα) is an immune inhibitory receptor on myeloid cells including macrophages and dendritic cells, which binds to CD47, a ubiquitous self-associated molecule. SIRPα-CD47 interaction is exploited by cancer cells to suppress anti-tumor activity of myeloid cells, therefore emerging as a novel immune checkpoint for cancer immunotherapy. In blood cancer, several SIRPα-CD47 blockers have shown encouraging monotherapy activity. However, the anti-tumor activity of SIRPα-CD47 blockers in solid tumors seems limited, suggesting the need for combination therapies to fully exploit the myeloid immune checkpoint in solid tumors. Here we tested whether combination of SIRPα-CD47 blocker with antibody-drug conjugate bearing a topoisomerase I inhibitor DXd (DXd-ADC) would enhance anti-tumor activity in solid tumors. To this end, DS-1103a, a newly developed anti-human SIRPα antibody (Ab), was assessed for the potential combination benefit with datopotamab deruxtecan (Dato-DXd) and trastuzumab deruxtecan (T-DXd), DXd-ADCs targeting human trophoblast cell-surface antigen 2 and human epidermal growth factor receptor 2, respectively. DS-1103a inhibited SIRPα-CD47 interaction and enhanced antibody-dependent cellular phagocytosis of Dato-DXd and T-DXd against human cancer cells. In a whole cancer cell vaccination model, vaccination with DXd-treated cancer cells led to activation of tumor-specific T cells when combined with an anti-mouse SIRPα (anti-mSIRPα) Ab, implying the benefit of combining DXd-ADCs with anti-SIRPα Ab on anti-tumor immunity. Furthermore, in syngeneic mouse models, both Dato-DXd and T-DXd combination with anti-mSIRPα Ab showed stronger anti-tumor activity over the monotherapies. Taken together, this study provides a preclinical rationale of novel therapies for solid tumors combining SIRPα-CD47 blockers with DXd-ADCs., Competing Interests: All authors are paid-employees of Daiichi Sankyo Co., Ltd. This does not alter our adherence to PLOS ONE policies on sharing data and materials., (Copyright: © 2024 Sue et al. This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.)

Recent advances in bleaching technology, such as bleaching ingredients, have accelerated the development of tooth whitening materials to change the color of natural teeth toward a lighter or whiter shade. Some bleaching materials are used for patients in combination with auxiliary medical devices to activate the bleaching materials by a light or heat source. Bleaching ingredients can be a poisonous and deleterious substance. Some bleaching materials are used in dental offices and the others at home. In clinical development of bleaching materials, appropriate evaluation of safety and efficacy is required according to the intended use. For appropriate and swift approval of the emerging bleaching materials, guideline on evaluation of dental whitening materials was discussed in a commissioned project by the Ministry of Health, Labour and Welfare of Japan for FY2021. Here, we summarize the current principles of the premarketing evaluation of the safety and efficacy., Competing Interests: The authors declare no conflict of interest., (© 2023 The Authors.)

Background: Mild cognitive impairment (MCI) is not just a prodrome to dementia, but a very important intervention point to prevent dementia caused by Alzheimer's disease (AD). It has long been known that people with AD have a higher frequency of falls with some gait instability. Recent evidence suggests that vestibular impairment is disproportionately prevalent among individuals with MCI and dementia due to AD. Therefore, we hypothesized that the measurement of balance capability is helpful to identify individuals with MCI., Methods: First, we developed a useful method to evaluate balance capability as well as vestibular function using Nintendo Wii balance board as a stabilometer and foam rubber on it. Then, 49 healthy volunteers aged from 56 to 75 with no clinically apparent cognitive impairment were recruited and the association between their balance capability and cognitive function was examined. Cognitive functions were assessed by MoCA, MMSE, CDR, and TMT-A and -B tests., Results: The new balance capability indicator, termed visual dependency index of postural stability (VPS), was highly associated with cognitive impairment assessed by MoCA, and the area under the receiver operating characteristic (ROC) curve was more than 0.8, demonstrating high sensitivity and specificity (app. 80% and 60%, respectively)., Conclusions: Early evidence suggests that VPS measured using Nintendo Wii balance board as a stabilometer helps identify individuals with MCI at an early and preclinical stage with high sensitivity, establishing a useful method to screen MCI., (© 2023. The Author(s).)

Adipose-derived stem cells (ASCs) are an attractive cell source for cell therapy. Despite the increasing number of clinical applications, the methodology for ASC isolation is not optimized for every individual. In this study, we developed an effective material to stabilize explant cultures from small-fragment adipose tissues., Methods: Polypropylene/polyethylene nonwoven sheets were coated with hydroxyapatite (HA) particles. Adipose fragments were then placed on these sheets, and their ability to trap tissue was monitored during explant culture. The yield and properties of the cells were compared to those of cells isolated by conventional collagenase digestion., Results: Hydroxyapatite-coated nonwovens immediately trapped adipose fragments when placed on the sheets. The adhesion was stable even in culture media, leading to cell migration and proliferation from the tissue along with the nonwoven fibers. A higher fiber density further enhanced cell growth. Although cells on nonwoven explants could not be fully collected with cell dissociation enzymes, the cell yield was significantly higher than that of conventional monolayer culture without impacting stem cell properties., Conclusions: Hydroxyapatite-coated nonwovens are useful for the effective primary explant culture of connective tissues without enzymatic cell dissociation., Competing Interests: The authors declare no conflicts of interest associated with this manuscript., (© 2022 The Japanese Society for Regenerative Medicine. Production and hosting by Elsevier B.V.)

Introduction: Cell therapy using adipose-derived mesenchymal stem cells (ASCs) is a promising avenue of regenerative medicine for the treatment of various diseases. It has been considered that ASCs exert their therapeutic effects through the secretion of multiple factors that are critical for tissue remodeling or the suppression of inflammation. Recently, conditioned medium (CM) from ASCs that contains a complex of secreted factors has received attention as a cost-effective alternative to cell therapy., Methods: We investigated the effects of CM obtained from ASCs (ASCs-CM) using human dermal fibroblasts (hDFs) and human epidermal keratinocytes with or without interleukin (IL)-1β and examined mRNA levels of marker genes. We also examined alterations in cell proliferation and morphology of hDFs following treatment with ASCs-CM. We further investigated the effects of ASCs-CM treatment on prevention of skin inflammation using a mouse model., Results: In hDFs and human epidermal keratinocytes, the ASCs-CM treatment suppressed pro-inflammatory factors and enhanced regenerative and remodeling factors with or without interleukin (IL)-1β exposure. The ASCs-CM treatment also enhanced cell proliferation of hDFs and prevented morphological changes in response to IL-1β exposure. Furthermore, in a mouse model of skin inflammation, treatment with ASCs-CM reduced the inflammatory reactions, including redness and thickness., Conclusions: CM from ASCs may represent a potential alternative to ASC therapy for the treatment of inflammatory skin conditions., Competing Interests: The authors declare that they have no conflicts of interest., (© 2022 The Japanese Society for Regenerative Medicine. Production and hosting by Elsevier B.V.)

Small antibody mimetics that contain high-affinity target-binding peptides can be lower cost alternatives to monoclonal antibodies (mAbs). We have recently developed a method to create small antibody mimetics called FLuctuation-regulated Affinity Proteins (FLAPs), which consist of a small protein scaffold with a structurally immobilized target-binding peptide. In this study, to further develop this method, we established a novel screening system for FLAPs called monoclonal antibody-guided peptide identification and engineering (MAGPIE), in which a mAb guides selection in two manners. First, antibody-guided design allows construction of a peptide library that is relatively small in size, but sufficient to identify high-affinity binders in a single selection round. Second, in antibody-guided screening, the fluorescently labeled mAb is used to select mammalian cells that display FLAP candidates with high affinity for the target using fluorescence-activated cell sorting. We demonstrate the reliability and efficacy of MAGPIE using daclizumab, a mAb against human interleukin-2 receptor alpha chain (CD25). Three FLAPs identified by MAGPIE bound CD25 with dissociation constants of approximately 30 nM as measured by biolayer interferometry without undergoing affinity maturation. MAGPIE can be broadly adapted to any mAb to develop small antibody mimetics., (© 2021. The Author(s).)

Rationale: Although capillary hemangiomas, common lesions involving the proliferation of small capillary vessels and a single layer of endothelial cells, can arise in any organ, they are rarely reported in the greater or lesser omentum. Here in, we report a case of capillary hemangioma arising from the lesser omentum in an adult with interesting diagnostic imaging findings, including changes in tumor size over time on computed tomography (CT), that was resected using laparoscopic surgery. To our knowledge, this is the first English report to describe a capillary hemangioma arising from the lesser omentum., Patient Concerns: A 63-year-old Japanese man received hemodialysis for chronic renal failure due to diabetic nephropathy, and a small, gradually enlarging tissue mass was found near the lesser curvature of the stomach on plain CT performed annually, without any associated complaints. Diagnostic imaging revealed an 18 × 15-mm tumor with a homogenous, highly enhanced effect in the early phase that was attenuated but prolonged in the delayed phase. Magnetic resonance imaging showed a mass with low signal intensity on T1-weighted imaging and relatively high signal intensity on T2-weighted imaging., Diagnosis: The patient was diagnosed with capillary hemangioma arising from the lesser omentum according to the pathological and immunohistological findings., Interventions: The patient underwent laparoscopy for excision of the tumor from the lesser omentum., Outcomes: At the 1 year follow-up, the patient had no recurrence of the tumor., Lessons: We describe the first case worldwide of capillary hemangioma that was a true vascular tumor arising from the lesser omentum. Although capillary hemangioma arising from the lesser omentum is extremely rare, it should be considered in the differential diagnosis of patients presenting with a highly enhanced lesser omental tumor, and laparoscopy can be safely applied for the excision of this tumor.

Background: Internal hernia is a rare cause of intestinal obstruction, and sigmoid mesocolon hernia is an extremely rare form of this condition. Among sigmoid mesocolon hernias, intramesosigmoid hernia is the least frequent subtype. We described a case of intramesosigmoid hernia through the orifice on the right leaf of the mesosigmoid with an incarcerated ileum of 6 cm in length without strangulation. This case was diagnosed by multidetector computed tomography with multiplanar reconstruction images and treated without resection of the small intestine in a 52-year-old man with characteristic diagnostic images., Case Presentation: A 52-year-old man suffering periumbilical cramping pain with sudden onset that had persisted for 1 week without recovery was referred to Fukui Katsuyama General Hospital. Multidetector computed tomography revealed small bowel obstruction, and an incarcerated short intestinal loop was revealed by sagittal slices of the multiplanar reconstruction images of the routine study of the left side of the pelvic space. Sagittal multiplanar reconstruction images also showed narrow belt-shaped fluid retention contacting the tip of the incarcerated short loop toward the cranial direction localized in the mesosigmoid. These findings indicated that the fluid and the herniated small bowel were wrapped together in the mesosigmoid, which was characteristic of intramesosigmoid hernia. The patient underwent laparotomy operation 2 days after admission. The ileum, which was approximately 75 cm proximal to the ileocecal junction and herniated into the mesosigmoid through the right leaf, was released without resection. The orifice located in the central part of the right leaf was oval shaped and measured less than 2 cm in diameter. The left leaf of the mesosigmoid was intact. The orifice of the right lobe was closed by suture. The patient showed an uneventful recovery., Conclusion: We report an extremely rare case of incarcerated intramesosigmoid hernia that was diagnosed by multidetector computed tomography with multiplanar reconstruction images. The finding of narrow belt-shaped fluid retention contacting the tip of the incarcerated short intestinal loop is characteristic of intramesosigmoid hernia and will be useful for conclusively differentiating this disease from transmesosigmoid hernia. Although intramesosigmoid hernia is a rare cause of internal hernia, multidetector computed tomography and multiplanar reconstruction images can provide the characteristic findings and proved useful for the precise preoperative diagnosis and treatment of intramesosigmoid hernia.

The immunosuppressive tumor microenvironment is a hallmark of cancer. Myeloid-derived suppressor cells (MDSCs) are CD11b + Gr-1 + tumor-infiltrating immature myeloid cells that strongly mediate tumor immunosuppression. The CD11b + Gr-1 + cells are a heterogeneous cell population, and the impacts of each subpopulation on tumor progression are not yet completely understood. In the present study, we identified a novel subpopulation of CD11b + Gr-1 + cells from murine lung carcinoma tumors according to their strongly adherent abilities. Although strong adherent activity is a unique property of macrophages, their marker expression patterns are similar to those of MDSCs; thus, we named this novel subpopulation MDSC-like adherent cells (MLACs). Unlike known MDSCs, MLACs lack the ability to suppress cytotoxic T lymphocytes and differentiate into tumor-associated macrophages (TAMs), but could still directly facilitate tumor growth and angiogenesis through secreting CCL2, CXCL1/2/5, PAI-1, MMPs, and VEGFA. Furthermore, MLACs recruited MDSCs via the secretion of CCL2/5 and CXCL1/2/5, thereby enhancing the immunosuppressive tumor microenvironment and promoting TAMs-mediated tumor progression. Our findings suggest that MLACs may function as an initiator of the immunosuppressive tumor microenvironment and highlight a new therapeutic target to prevent the onset or delay malignant progression., Competing Interests: CONFLICTS OF INTEREST The authors declare no conflicts of interest.

Vasohibins (VASH1 and VASH2) are recently identified regulators of angiogenesis and cancer cell functions. They are secreted proteins without any classical secretion signal sequences, and are thought to be secreted instead via an unconventional protein secretion (UPS) pathway in a small vasohibin-binding protein (SVBP)-dependent manner. However, the precise mechanism of SVBP-dependent UPS is poorly understood. In this study, we identified a novel UPS regulatory system in which essential domain architecture (VASH-PS) of VASHs, comprising regions VASH1 91-180 and VASH2 80-169 , regulate the cytosolic punctate structure formation in the absence of SVBP. We also demonstrate that SVBP form a complex with VASH1 through the VASH1 274-282 (SIa), VASH1 139-144 (SIb), and VASH1 133-137 (SIc), leading to the dispersion in the cytosol and extracellular release of VASH1. The amino acid sequences of VASH-SIa and VASH-PS, containing SIb and SIc, are highly conserved among VASH family members in vertebrates, suggesting that SVBP-dependent UPS may be common within the VASH family. This novel UPS regulatory system may open up new avenues for understanding fundamental protein secretion in vertebrates., (© 2016 The Protein Society.)

Background: Leukotriene receptor antagonists have been used to prevent virus-induced asthma exacerbations in autumn. Its efficacy, however, might differ with age and sex., Objective: This study aimed to investigate whether pranlukast added to usual asthma therapy in Japanese children during autumn, season associated with the peak of asthma, reduces asthma exacerbations. It was also evaluated the effect of age and sex on pranlukast's efficacy., Methods: A total of 121 asthmatic children aged 1 to 14 years were randomly assigned to receive regular pranlukast or not according to sex, and were divided in 2 age groups, 1-5 years and 6-14 years. The primary outcome was total asthma score calculated during 8 weeks by using a sticker calendar related to the days in which a child experienced a worsening of asthma symptoms. This open study lasted 60 days from September 15 to November 14, 2007., Results: Significant differences in pranlukast efficacy were observed between sex and age groups. Boys aged 1 to 5 years had the lower total asthma score at 8 weeks ( p = 0.002), and experienced fewer cold episodes ( p = 0.007). There were no significant differences between pranlukast and control group in total asthma score at 8 weeks ( p = 0.35), and in the days in which a child experienced a worsening of asthma symptoms ( p = 0.67)., Conclusion: There was a substantial benefit of adding pranlukast to usual therapy in asthmatic children, especially in boys aged 1 to 5 years, during autumn season.

Peptides that have high affinity for target molecules on the surface of cancer cells are crucial for the development of targeted cancer therapies. However, unstructured peptides often fail to bind their target molecules with high affinity. To efficiently identify high-affinity target-binding peptides, we have constructed a fluorescent protein scaffold, designated gFPS, in which structurally constrained peptides are integrated at residues K131-L137 of superfolder green fluorescent protein. Molecular dynamics simulation supported the suitability of this site for presentation of exogenous peptides with a constrained structure. gFPS can present 4 to 12 exogenous amino acids without a loss of fluorescence. When gFPSs presenting human epidermal growth factor receptor type 2 (HER2)-targeting peptides were added to the culture medium of HER2-expressing cells, we could easily identify the peptides with high HER2-affinity and -specificity based on gFPS fluorescence. In addition, gFPS could be expressed on the yeast cell surface and applied for a high-throughput screening. These results demonstrate that gFPS has the potential to serve as a powerful tool to improve screening of structurally constrained peptides that have a high target affinity, and suggest that it could expedite the one-step identification of clinically applicable cancer cell-binding peptides.

Enzyme-linked immunosorbent assays (ELISA) have been widely used to determine quantitatively autoantibodies. However, the processes for the purification and immobilization of antigens in conventional ELISA methods include multiple steps, which have hampered the application for screening of autoantibodies. Here, we have developed a novel ELISA system using the plates pre-coated with glutathione casein to capture recombinant proteins fused to N-terminal glutathione S-transferase (GST). The GST-fused proteins were synthesized with the wheat germ cell-free protein production system. Thus, the present system combined the GST-capture ELISA with the cell-free protein production system, which allowed immobilization of the recombinant proteins with one-step purification. Using this ELISA method, we determined whether rheumatoid factors (RF), which have been considered as one of the representative disease-specific autoantibodies for rheumatoid arthritis (RA), were genetically associated with severity of arthritis in a mouse model for RA, MRL/Mp-lpr/lpr (MRL/lpr). GST-fused human IgG1-Fc (GST-Fc), synthesized with the robotic protein synthesizer, were used as reactants for RF. Serum samples for RF were prepared from 11 lines of a recombinant inbred mouse strain, MXH/lpr, which was established from intercrosses between MRL/lpr and non-arthritic C3H/HeJ-lpr/lpr (C3H/lpr) strains, composed of a different genomic recombination derived from the parental strains in each line. A correlation of RF titers with the severity of the arthritis in these lines was not significant, indicating genetic dissociation of RF from arthritis and that RF is not necessarily required for the development of RA. The present method may provide high-throughput screening for determining the disease-specific autoantibodies in autoimmune diseases.

Objective: Crescent formation in the renal glomerulus is a typical manifestation of progressive glomerulopathy associated with fatal renal failure; therefore, its prevention is of clinical importance. Little is known about the pathogenic mechanism for crescent formation. This study was undertaken in an attempt to identify the events that are critical for crescent formation in immune complex crescentic glomerulonephritis (CGN) by analyzing a novel mutant strain of mice., Methods: A spontaneous mutant strain of mice was isolated from the autoimmune-prone strain EOD, which stably develops fatal CGN. The mutant phenotypes were assessed histopathologically, hematologically, and immunologically. The mutation was searched for with positional cloning using microsatellite markers., Results: Compared with wild-type EOD (WT-EOD) mice, mutant EOD (mut-EOD) mice showed marked improvement in CGN in conjunction with an improvement in spontaneous mortality. In WT-EOD mice, an inverse correlation between blood urea nitrogen concentration and blood platelet count and massive accumulation of platelets in the glomerulus were evident, suggesting that an accumulation of platelets in the glomerulus contributes to the progression of CGN. The mutant platelets showed an abnormal aggregation in response to collagen and thrombin, associated with a bleeding tendency in mut-EOD mice. Genetic analysis revealed a deleterious mutation in the cappuccino gene (cno), which encodes a protein that belongs to a complex called the biogenesis of lysosome-related organelle complex 1 and is profoundly involved in platelet function. Morphologic examination revealed a partial defect in dense body formation in the delta-granule of platelets., Conclusion: The present findings suggest that platelet functions have a critical role in crescent formation in autoimmune GN.