Primary hyperparathyroidism (PHPT) typically results from parathyroid adenoma, multiglandular hyperplasia, or parathyroid carcinoma. Patients usually present with skeletal manifestations such as low-trauma fractures. Osteitis fibrosa cystica (OFC) is a classic yet rare skeletal manifestation of advanced PHPT currently reported in less than 2% of patients. We present a case of a 29-year-old Indian female who presented with a femur fracture and mandibular OFC 20 days after delivery. The painless mandibular swelling gradually progressed from the third month of pregnancy. The biochemical and radiological investigations were indicative of PHPT-associated OFC. After the excision of the three-and-a-half parathyroid gland, histology revealed benign cystic adenomas and hyperplasia. Based on the associated clinical manifestations, OFC was suspected. Clinical exome sequencing revealed CDC73(+) c.687_688dupAG heterogenous pathogenic autosomal dominant mutation. Undiagnosed PHPT in mothers during pregnancy led to neonatal hypocalcaemic convulsions. With adequate supplementation, the infant recovered completely from transient congenital hypoparathyroidism. OFC is an important diagnosis to consider in a young patient with swelling of the neck and jaw. Simultaneous high levels of PTH and serum calcium should raise a high index of suspicion for OFC. Parathyroidectomy helps manage the biochemical abnormalities and causes regression of the jaw mass that causes facial disfigurement and attenuates the declining BMD. Children born to mothers with PHPT should be evaluated for neonatal hypoparathyroidism and supplemented appropriately to reduce the risk of hypocalcaemic manifestations that can be life-threatening. If the CDC73 mutation is detected, the offspring should be monitored for signs of PHPT due to the high probability of inheritance and parathyroid malignancy.
Osteitis Fibrosa Cystica ; Hyperparathyroidism, Primary ; Fracture, Pathological ; Fractures, Spontaneous

In this paper, we review the results of previous studies and summarize the effects of various factors on the regulation of bone metabolism in traumatic bone infections. Infection-related bone destruction incorporates pathogens and iatrogenic factors in the process of bone resorption dominated by the skeletal and immune systems. The development of bone immunology has established a bridge of communication between the skeletal system and the immune system. Exploring the effects of pathogens, skeletal systems, immune systems, and antibacterials on bone repair in infectious conditions can help improve the treatment of these diseases.
Anti-Bacterial Agents/administration & dosage* ; Bone and Bones/metabolism* ; Cellular Microenvironment ; Humans ; Immune System/immunology* ; Lymphocyte Subsets/immunology* ; Osteitis/microbiology* ; Osteoblasts/physiology* ; Osteoclasts/physiology* ; Staphylococcal Infections

PURPOSE: Osteitis refers to the development of new bone formation and remodeling of bone in chronic rhinosinusitis (CRS) patients; it is typically associated with eosinophilia, nasal polyps (NPs), and recalcitrant CRS. However, the roles of ossification in CRS with or without NPs remain unclear due to the lack of appropriate animal models. Thus, it is necessary to have a suitable animal model for greater advances in the understanding of CRS pathogenesis.METHODS: BALB/c mice were administered ovalbumin (OVA) and staphylococcal enterotoxin B (SEB). The numbers of osteoclasts and osteoblasts and bony changes were assessed. Micro computed tomography (micro-CT) scans were conducted to measure bone thickness. Immunofluorescence, immunohistochemistry, and quantitative polymerase chain reaction were performed to evaluate runt-related transcription factor 2 (RUNX2), osteonectin, interleukin (IL)-13, and RUNX2 downstream gene expression. Gene set enrichment analysis was performed in mucosal tissues from control and CRS patients. The effect of resveratrol was evaluated in terms of osteogenesis in a murine eosinophilic CRS NP model.RESULTS: The histopathologic changes showed markedly thickened bones with significant increase in osteoblast numbers in OVA/SEB-treated mice compared to the phosphate-buffered saline-treated mice. The structural changes in bone on micro-CT were consistent with the histopathological features. The expression of RUNX2 and IL-13 was increased by the administration of OVA/SEB and showed a positive correlation. RUNX2 expression mainly co-localized with osteoblasts. Bioinformatic analysis using human CRS transcriptome revealed that IL-13-induced bony changes via RUNX2. Treatment with resveratrol, a candidate drug against osteitis, diminished the expression of IL-13 and RUNX2, and the number of osteoblasts in OVA/SEB-treated mice.CONCLUSIONS: In the present study, we found the histopathological and radiographic evidence of osteogenesis using a previously established murine eosinophilic CRS NP model. This animal model could provide new insights into the pathophysiology of neo-osteogenesis and provide a basis for developing new therapeutics.
Animals ; Computational Biology ; Core Binding Factor Alpha 1 Subunit ; Enterotoxins ; Eosinophilia ; Eosinophils ; Fluorescent Antibody Technique ; Gene Expression ; Humans ; Immunohistochemistry ; Interleukin-13 ; Interleukins ; Mice ; Models, Animal ; Mucous Membrane ; Nasal Polyps ; Nose ; Osteitis ; Osteoblasts ; Osteoclasts ; Osteogenesis ; Osteonectin ; Ovalbumin ; Polymerase Chain Reaction ; Sinusitis ; Transcription Factors ; Transcriptome

BACKGROUND: Mycobacterium bovis Bacille Calmette-Guérin (BCG) osteitis, a rare complication of BCG vaccination, has not been well investigated in Korea. This study aimed to evaluate the clinical characteristics of BCG osteitis during the recent 10 years in Korea. METHODS: Children diagnosed with BCG osteitis at the Seoul National University Children's Hospital from January 2007 to March 2018 were included. M. bovis BCG was confirmed by multiplex polymerase chain reaction (PCR) in the affected bone. BCG immunization status and clinical information were reviewed retrospectively. RESULTS: Twenty-one patients were diagnosed with BCG osteitis and their median symptom onset from BCG vaccination was 13.8 months (range, 6.0–32.5). Sixteen children (76.2%) received Tokyo-172 vaccine by percutaneous multiple puncture method, while four (19.0%) and one (4.8%) received intradermal Tokyo-172 and Danish strain, respectively. Common presenting symptoms were swelling (76.2%), limited movement of the affected site (63.2%), and pain (61.9%) while fever was only accompanied in 19.0%. Femur (33.3%) and the tarsal bones (23.8%) were the most frequently involved sites; and demarcated osteolytic lesions (63.1%) and cortical breakages (42.1%) were observed on plain radiographs. Surgical drainage was performed in 90.5%, and 33.3% of them required repeated surgical interventions due to persistent symptoms. Antituberculosis medications were administered for a median duration of 12 months (range, 12–31). Most patients recovered without evident sequelae. CONCLUSION: Highly suspecting BCG osteitis based on clinical manifestations is important for prompt management. A comprehensive national surveillance system is needed to understand the exact incidence of serious adverse reactions following BCG vaccination and establish safe vaccination policy in Korea.
Child ; Drainage ; Femur ; Fever ; Humans ; Immunization* ; Incidence ; Korea* ; Methods ; Multiplex Polymerase Chain Reaction ; Mycobacterium bovis* ; Mycobacterium* ; Osteitis* ; Punctures ; Retrospective Studies ; Seoul ; Tarsal Bones ; Vaccination

PURPOSE: To evaluate the Japanese Epidemiological Survey of Refractory Eosinophilic Chronic Rhinosinusitis (JESREC) classification, a clinical scoring system, for predicting disease control status in chronic rhinosinusitis with nasal polyps (CRSwNP) and to investigate prognostic factors. MATERIALS AND METHODS: In total, 134 CRSwNP patients who underwent functional endoscopic sinus surgery after maximal medical treatment were enrolled. These patients were categorized into four groups according to JESREC classification: 1) non-eosinophilic CRSwNP (non-ECRSwNP), 2) mild eosinophilic CRSwNP (ECRSwNP), 3) moderate ECRSwNP, and 4) severe ECRSwNP. Disease control status among the patients was evaluated at 1 year after surgery, and the patients were divided into two groups (disease-controlled and disease-uncontrolled groups) for the investigation of prognostic factors. RESULTS: There was no significant difference in disease control status between non-ECRSwNP and ECRSwNP groups (p=0.970). Age, Lund-Mackay CT scores, global osteitis scores, tissue neutrophil count, and tissue eosinophil count were associated with disease control status. In subgroup analysis of the non-ECRSwNP group, only high tissue neutrophil count was related with disease control status, whereas for the ECRSwNP group, young age, high Lund-Mackay CT scores, high global osteitis scores, and high tissue and blood eosinophil counts were associated with disease control status. CONCLUSION: No difference in disease control status was identified between non-ECRSwNP and ECRSwNP cases. Tissue neutrophilia, however, appeared to be associated with disease control status in non-ECRSwNP cases, whereas tissue and blood eosinophilia was associated with ECRSwNP cases.
Asian Continental Ancestry Group ; Classification ; Eosinophilia ; Eosinophils ; Humans ; Nasal Polyps ; Neutrophils ; Osteitis ; Prognosis ; Sinusitis

SAPHO (synovitis, acne, pustulosis, hyperostosis, and osteitis) syndrome includes a variety of inflammatory bone disorders associated with dermatologic pathology. A 57-year-old female presented with pustulosis on both hands that had persisted for several months. She also had lower back pain without trauma history. On physical examination, tenderness on her lower back and left anterior chest wall pain were found, and claudication was observed. Radiological studies including computed tomography (CT), magnetic resonance imaging (MRI), and positron emission tomography (PET)-CT showed endplate lytic changes in her spine, a focal hypermetabolic lesion in a left rib, and costochondral junction. These findings raised doubt on the presence of metastatic bone lesions, and there was no indication for primary cancer after a complete medical checkup. Palmoplantar pustulosis was well controlled by treatment with acitretin. The osteitis associated with SAPHO syndrome usually presents as osteosclerosis, while reports on osteolytic lesions are rare. We report herein a rare case of SAPHO syndrome associated with bone lesions resembling bone metastasis.
Acitretin ; Acne Vulgaris ; Acquired Hyperostosis Syndrome ; Female ; Hand ; Humans ; Hyperostosis ; Low Back Pain ; Magnetic Resonance Imaging ; Middle Aged ; Neoplasm Metastasis ; Osteitis ; Osteosclerosis ; Pathology ; Physical Examination ; Positron-Emission Tomography ; Ribs ; Spine ; Thoracic Wall

Pustulotic arthro-osteitis (PAO) is a rare chronic inflammatory disorder characterized by inflammatory osteitis of the sternoclavicular joint and palmoplantar pustulosis. Here, we report a case of PAO that was successfully treated with a TNF-α inhibitor. A 45-year-old man presented with a 3-month history of pustular eruption on the palms and soles. Physical examination showed multiple erythematous papulopustules on the palms, back, and left shin, accompanied by sternoclavicular joint swelling and tenderness. Skin biopsy showed intraepidermal pustules filled with neutrophils on the palm. Bone scintigraphy revealed increased uptake in the bilateral sternoclavicular and other axial joints. Based on these findings, we made the diagnosis of PAO. Even after 6-month treatment of oral steroids and cyclosporine, skin manifestations insufficiently improved, so etanercept therapy was started. Complete clearance of skin lesions and joint pain were achieved after 3 months of etanercept therapy.
Arthralgia ; Biopsy ; Cyclosporine ; Diagnosis ; Etanercept ; Humans ; Joints ; Middle Aged ; Neutrophils ; Osteitis ; Physical Examination ; Radionuclide Imaging ; Skin ; Skin Manifestations ; Sternoclavicular Joint ; Steroids

Alkaline Phosphatase ; metabolism ; Asian Continental Ancestry Group ; Bone Density Conservation Agents ; therapeutic use ; China ; Denosumab ; therapeutic use ; Diabetes Mellitus, Type 2 ; complications ; Female ; Humans ; Hyperlipidemias ; complications ; Hypertension ; complications ; Middle Aged ; Osteitis Deformans ; complications ; diagnostic imaging ; drug therapy ; metabolism ; Pelvic Bones ; diagnostic imaging ; Renal Insufficiency, Chronic ; complications ; Singapore ; Tibia ; diagnostic imaging ; Treatment Outcome

The causes of osteolytic lesions found in radiological examinations are not quite certain. Therefore, to determine the appropriate treatment method, various approaches and analyzes are required to find the real cause. Hyperparathyroidism is one of the diseases which forms osteolytic bone lesions so-called brown tumor. A 55-year-old woman who had painful osteolytic bone lesions in both hip joint areas was diagnosed as parathyroid carcinoma after serial work-up. She underwent parathyroidectomy and follow-up imaging showed a decrease in brown tumor size and bone consolidation in the subchondral bone destruction area. Proper evaluation of osteolytic bone lesions helps to avoid unnecessary operative treatments and the first choice for the treatment of osteolytic bone lesions caused by parathyroid carcinoma is parathyroidectomy.
Female ; Follow-Up Studies ; Hip Joint ; Humans ; Hyperparathyroidism ; Methods ; Middle Aged ; Osteitis Fibrosa Cystica ; Parathyroid Neoplasms ; Parathyroidectomy

Dry socket, also termed fibrinolytic osteitis or alveolar osteitis, is a complication of tooth exodontia. A dry socket lesion is a post-extraction socket that exhibits exposed bone that is not covered by a blood clot or healing epithelium and exists inside or around the perimeter of the socket or alveolus for days after the extraction procedure. This article describes dry socket lesions; reviews the basic clinical techniques of treating different manifestations of dry socket lesions; and shows how microscope level loupe magnification of 6× to 8× or greater, combined with co-axial illumination or a dental operating microscope, facilitate more precise treatment of dry socket lesions. The author examines the scientific validity of the proposed causes of dry socket lesions (such as bacteria, inflammation, fibrinolysis, or traumatic extractions) and the scientific validity of different terminologies used to describe dry socket lesions. This article also presents an alternative model of what causes dry socket lesions, based on evidence from dental literature. Although the clinical techniques for treating dry socket lesions seem empirically correct, more evidence is required to determine the causes of dry socket lesions.
Bacteria ; Diagnosis* ; Dry Socket* ; Epithelium ; Fibrinolysis ; Inflammation ; Lighting ; Osteitis ; Tooth