Bone development and lung function are integral to child and adolescent health. Both influence an individual\'s overall well-being and potentially affect long-term health. Utilizing a comprehensive dataset from the National Health and Nutrition Examination Survey, this study aims to elucidate the relationship between lung function and bone mineral density (BMD) in a representative sample of children and adolescents. The analysis covered 3410 participants aged 8 to 19 years. We employed weighted multivariate linear regression and restricted cubic spline curve visualizations to explore the intricate association between lung function metrics, particularly first-second expiratory volume 1 second/forced vital capacity ratio, and lumbar BMD. Our data indicated a positive association between lung function and lumbar BMD in children and adolescents. Specifically, higher lung function metrics were linked with increased lumbar BMD. This association was more pronounced in younger participants or those with a lower body mass index. A significant positive relationship exists between lung function and BMD in the pediatric population. Recognizing this association is crucial for holistic health strategies for children and adolescents. This study underscores the need for integrated health monitoring during formative years, which can influence health trajectories as these individuals transition to adulthood.

Family with sequence similarity 20-member C (FAM20C) is a kinase specific to most of the secreted phosphoproteome. FAM20C has been identified as the causative gene of Raine syndrome, initially characterized by lethal osteosclerosis bone dysplasia. However, since the identification of the cases of nonlethal Raine syndrome characterized by hypophosphatemia rickets, the previous definition of Raine syndrome has become debatable and raised a question about the role of mutations of FAM20C in controversial skeletal manifestation in the two forms of the disease. In this study, we aimed to investigate the influence of FAM20C mutations on skeletogenesis. We developed transgenic mice expressing Fam20c mutations mimicking those associated with human lethal and nonlethal Raine syndrome. The results revealed that transgenic mice expressing the mutant Fam20c found in the lethal (KO;G374R) and nonlethal (KO;D446N) Raine syndrome exhibited osteomalacia without osteosclerotic features. Additionally, both mutants significantly increased the expression of the Fgf23, indicating that Fam20c deficiency in skeletal compartments causes hypophosphatemia rickets. Furthermore, as FAM20C kinase activity catalyzes the phosphorylation of secreted proteomes other than those in the skeletal system, global FAM20C deficiency may trigger alterations in other systems resulting in osteosclerosis secondary to hypophosphatemia rickets. Together, the findings of this study suggest that FAM20C deficiency primarily causes hypophosphatemia rickets or osteomalacia; however, the heterogeneous skeletal manifestation in Raine syndrome was not determined solely by specific mutations of FAM20C. The findings also implicated that rickets or osteomalacia caused by FAM20C deficiency would deteriorate into osteosclerosis by the defects from other systems or environmental impacts.

To study the effects of low-density lipoprotein receptor-related protein 5 (LRP5) gene mutations on bone, and to open up our view of LRP5 and Wnt pathways on bone mass regulation. Three patients with increased bone mineral density or thickened bone cortex were included, who were 30-year-old, 22-year-old and 50-year-old men, respectively. The latter two patients were son and father of a same family. The characteristics of bone X-rays were evaluated in detail. Bone turnover markers were detected, such as procollagen type 1 amino-terminal peptide (P1NP), alkaline phosphatase (ALP), and type 1 collagen carboxyl terminal peptide (β-CTX). Dual energy X-ray absorptiometry (DXA) was used to measure the bone mineral density (BMD) at lumbar spine and proximal femur of the patients. The targeted next-generation sequencing (NGS) technology was used to detect pathogenic gene mutations, which were further verified by Sanger sequencing. Moreover, the gene mutation spectrum and phenotypic characteristics of reported patients with LRP5 gain-of-function mutations were summarized by reviewing the literature. The main characteristics of the first patient were headache, facial paralysis, high BMD (lumbar vertebrae 1-4: 1.877 g/cm2, Z-score: 5.8; total hip: 1.705 g/cm2, Z-score: 5.7), slightly increased P1NP (87.0 ng/mL) and β-CTX (0.761 ng/mL) level, and with thickened bone cortex, especially the cranial vault. The latter two patients showed enlargement of the mandible and enlarged osseous prominence of the tours palatinus. X-rays showed that the bone cortex of skull and long bones were thickened. The bone turnover markers and BMD were normal. All three cases carried novel missense mutations in LRP5 gene, which were mutation in exon 3 (c.586 T > G, p.Trp196Gly) of the first patient, and mutation in exon 20 (c.4240C > A, p.Arg1414Ser) of the latter two patients. Combined with the reported literature, a total of 19 gain-of-function mutations in LRP5 were detected in 113 patients from 33 families. Hotspot mutations included c.724G > A, c.512G > T and c.758C > T. Furthermore, mutations in the exon 3 of LRP5 may cause severe phenotypes. LRP5 gain-of-function mutations can lead to rare autosomal dominant osteosclerosis type Ι (ADO Ι), which was characterized by increased bone mass and thickened bone cortex. In-depth research on the Wnt pathway will be benefit for discovering important mechanisms of bone mass regulation.

Fluoride (F) exposure can cause osteosclerosis, which is characterised by a high bone mass, but its mechanism is not fully illustrated. Here, we aimed to evaluate the effects of excessive F exposure on the bone lesion by treating female Sprague-Dawley rats with different concentrations of sodium fluoride (NaF) (0, 55, 110 and 221 mg/L) for 90 days and the corresponding concentrations of fluorine ion (0, 25, 50 and 100 mg/L, respectively). Histopathological results showed that excessive F exposure caused the enlargement of trabeculae and their integration into one large piece, growth plate thickening, articular cartilage impairment and bone collagen abnormality. Meanwhile, F promoted calcium deposition and bone mineralisation, and induced abnormal osteogenesis increased. The results of micro-computed tomography also confirmed that excessive F destroyed the bone microstructure and induced a high-bone-mass phenotype, consistent with the results of pathomorphology. Mechanistically, excessive amounts of F led to angiogenesis inhibition and HIF-1α signalling enhancement. Subsequently, F induced autophagy and canonical Wnt/β-catenin signalling pathway activation. Collectively, these results manifested that F enhanced the hypoxia inducible factor-1α signalling, which in turn triggered autophagy and canonical Wnt/β-catenin signalling activation, ultimately leading to osteosclerosis in the rats.

BACKGROUND: The neuroendocrine tumor (NET) is rare, accounting for about 0.5% of all tumors. NETs have the characteristics of metastasis, especially lymph nodes, liver, spleen, and bone.
METHODS: We report a 30-year-old man diagnosed with a NET with bone metastasis and presented with waist and leg pain. The imaging findings of this case showed multiple osteosclerosis and no apparent bone destruction. We collected the patient\'s previous examinations, including laboratory, imaging, and pathological examination to get a precise diagnosis. Given this case, we carried out symptomatic support treatment to relieve the patients\' pain symptoms.
CONCLUSIONS: Bone metastases from NETs of unknown primary site are rare in both clinical and imaging manifestations. The disease is mainly manifested as multiple osteosclerosis, accompanied by muscle soreness and pain. It is recommended to try chemotherapy for this disorder.

BACKGROUND: Osteoarthritis (OA) is the most common musculoskeletal disease, and it has a complex pathology and unknown pathogenesis. Chondrocyte ferroptosis is closely associated with the development of OA. As a common drug administered for the treatment of type 2 diabetes, metformin (Met) is known to inhibit the development of ferroptosis. However, its therapeutic effect in OA remains unknown. The present study aimed to explore the effects of Met on cartilage and subchondral bone in a mouse OA model and to explore the potential underlying mechanisms.
METHODS: A mouse OA model was induced using destabilization of the medial meniscus (DMM) surgery, chondrocyte ferroptosis was induced using an intra-articular injection of Erastin, and Met (200 mg/kg/day) was intragastrically administered for 8 weeks after surgery. H&E and Safranin O‑fast green staining were used to evaluate cartilage degeneration, and μ‑computed tomography was used to evaluate changes in subchondral bone microarchitecture. Moreover, immunohistochemical staining was performed to detect mechanistic metalloproteinases 13, type II collagen, glutathione peroxidase 4, acyl-CoA synthetase long-chain family member 4, solute carrier family 7 member 11 and p53. Runt-associated transcription factor 2 and CD31 were detected using immunofluorescent staining.
RESULTS: Met protected articular cartilage and reversed the abnormal expression of ferroptosis-related proteins in the chondrocytes of DMM mice. Moreover, intra-articular injection of Erastin induced ferroptosis in mouse chondrocytes, and Met eliminated the ferroptosis effects induced by Erastin and protected articular cartilage. In addition, the results of the present study demonstrated that Met alleviated the microstructural changes of subchondral osteosclerosis and reduced heterotypic angiogenesis in DMM mice.
CONCLUSIONS: Met alleviates the pathological changes of OA by inhibiting ferroptosis in OA chondrocytes, alleviating subchondral sclerosis and reducing abnormal angiogenesis in subchondral bone in advanced OA.

BACKGROUND: POEMS (polyneuropathy, organomegaly, endocrinopathy, M protein, and skin changes) syndrome is a rare and complicated disease related to multiple organs and systems. Here, we report a case of systemic mastocytosis (SM) that was misdiagnosed as a POEMS syndrome.
METHODS: A 42-year-old man presented with skin changes, diarrhea, and limb numbness.
METHODS: Positron emission tomography/computed tomography revealed extravascular volume overload, organomegaly, lymphadenopathy, and bone lesions with mixed lesions of osteosclerosis and osteolysis. Therefore, POEMS syndrome was suspected. Further histopathological and immunohistochemical examination of the bone marrow, lymph nodes, and gastric mucosa suggested a diagnosis of mastocytosis. The c-Kit D816V mutation confirmed the diagnosis of SM.
METHODS: The patient received the treatment of pegylated interferon-alpha weekly and glucocorticoid daily.
RESULTS: The symptoms relieved significantly.
CONCLUSIONS: There are many similar features between POEMS syndrome and SM, probably leading to misdiagnosis. This study analyzed the different points between them which can provide help for differentiation.

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The UV/monochloramine (UV/NH2Cl) process has attracted increasing attention in water treatment, in which hydroxyl radicals (HO•), reactive chlorine species (RCS) and reactive nitrogen species (RNS) are produced. This study investigated the effects of water matrices including halides, natural organic matter (NOM), alkalinity and pH, on the degradation kinetic of a variety of micropollutants and radical chemistry in the UV/NH2Cl process. The presence of chloride blunted HO• and Cl• impacts, but enhanced Cl2•- effect on micropollutants reactive toward Cl2•-. The presence of 30 μM bromide led to an 82% decrease in the specific pseudo-first-order rate constants (k\') by HO• (kHO•\'), and significantly diminished RCS efficacy. Reactive bromine species (RBS) were formed in the presence of bromide, while the contribution could not compensate for the decrease of HO• and RCS due to their lower reactivity toward micropollutants. Iodide rapidly transformed to HOI via reacting with NH2Cl, which resulted in a 59% decrease of kHO•\' and 12% ∼ 100% decreases of k\' by reactive halogen species (RHS) and RNS (kRHS + RNS\') for most micropollutants. Nevertheless, k\' of phenolic compounds, such as paracetamol, bisphenol A and salbutamol, increased in the presence of iodide by 78%, 360% and 130%, respectively, due to the roles of HOI and reactive iodine species (RIS). Bicarbonate decreased the contributions of HO• and RCS, but enhanced that of CO3•- for micropollutants reactive toward CO3•-. The presence of 1 mg/L NOM scavenged over half the amount of HO•, and also consumed RCS and RNS, resulting in significantly decreased removal of micropollutants. High pH value witnessed enhanced degradation for those micropollutants reactive toward RCS and RNS through deprotonation. The degradation of most micropollutants was inhibited in real drinking water and in the coexistence of halides. This study provides a better understanding of radical chemistry in the UV/NH2Cl process under a practical water treatment condition.

Radiographically assisted dental identification is an important means for individual identification. Specific identifiers help to quickly filter some of the possible corresponding AM and PM images at the beginning. The study seeks specific oral and maxillofacial identifiers in panoramic radiographs. A total of 920 panoramic radiographs from 460 live patients were used. The most recent radiograph served as the surrogate post-mortem (PM) record of an unidentified person, and the earliest radiograph served as the ante-mortem (AM) record of the same person. We evaluated the following four groups of identifiers of the images: (1) dental morphology, tooth number, and position; (2) dental treatment and pathology; (3) morphological identifiers of the jaw; and (4) pathological identifiers of the jaw. The ratio of each identifier being identified simultaneously in the AM and PM databases was determined. Specific identifiers were defined as those that appeared at low frequency (ratio: 0%-0.250%). A total of 18 specific oral and maxillofacial identifiers were determined. The specific identifiers were a retained deciduous tooth (0.011%), S-shaped deflection of a tooth root (0.012%), distal deflection of tooth root (0.017%), inverted impaction (0.018%), malposition (0.038%), supernumerary teeth (0.061%), mesial deflection of tooth root (0.092%), microdontia (0.136%), buccal/lingual impaction (0.188%), cementoma (0.002%), hypercementosis (0.002%), continuous crown (0.004%), pulp calcification (0.023%), attrition (0.030%), residual root (0.106%), root resorption (0.137%), implant (0.156%), and osteomyelitis (0.002%). Identifiers of the teeth and jaw can be used for human identification, and dental identifiers are more specific than identifiers of jaw.