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An adult, female, mixed breed sow from a commercial breeding unit, with a clinical history of lethargy, poor body condition, hypersalivation, dyspnoea and reddened ears, was submitted for necropsy. Post-mortem examination identified the presence of an intestinal volvulus, which developed around an osseous intra-abdominal mesenteric lesion. Gross pathology and histopathology combined with post-mortem computed tomography (CT) of the mass confirmed the lesion as intra-abdominal heterotopic mesenteric ossification (IHMO), according to criteria applied to human disease. This report highlights a rare case of porcine IHMO, a poorly understood metaplastic condition, comparable to the human disease. We highlight the importance of a combination of CT and histopathology to reach a final diagnosis of the disease. Histopathology and further investigation of similar cases may contribute to a better understanding of the pathogenesis in animals, which is currently poorly understood.
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We present two pediatric cases of nontraumatic myositis ossificans (MO), which initially mimicked infections, leading to unnecessary treatments. The first case involves an 11-year-old boy with acute left hip pain and swelling, misdiagnosed as a hip joint infection and treated with antibiotics before histology confirmed MO. The second case is a two-year-old girl who presented with limping and restricted hip movement, initially suspected to have septic arthritis. Following MRI and clinical reassessment, her condition was diagnosed as MO. Both cases highlight the challenges of diagnosing MO in children and underscore the importance of including it in differential diagnoses for suspected infections.
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Introduction: Myositis ossificans (MO) is the most frequent type of heterotopic ossification, occurring in individuals aged 20-40 years within major muscles, and roughly 75% of cases are triggered by trauma. It is most commonly present in locations with a high risk of injury, such as the quadriceps femoris and the arm's flexor muscles, and it is infrequently found in intra-articular sites. Case Report: A 19-year-old male with a history of pain, swelling, and restriction of movements of the right elbow for the last 6 years with a history of difficulty in eating and combing hair with the right hand. History of trauma to his right elbow 6 years ago for which he underwent conservative native treatment with massage. Physical examination found a swelling over the right medial elbow joint of size approximately 7 cm × 6 cm × 3 cm, with no local rise of temperature or tenderness. There was a fixed flexion deformity of 75°. There were no sensory or vascular deficits. Under general anesthesia, through the anterior approach, an irregular bony mass was visualized which was excised. Through a posterior approach, a skin incision was made, subcutaneous tissue dissected, muscles retracted and then a calcified mass visualized over the olecranon process which was excised. The post-operative period was uneventful. Elbow joint mobilization was started from day 2 which showed significant correction of fixed flexion deformity. Conclusion: It is necessary to rule out non-traumatic MO when a patient exhibits radiologically detectable calcification and complains of a painful lump forming in a muscle. The degree of calcification and mature bone growth varies depending on the stage of the lesion. A biopsy of the lesion can rule out malignant transformation. Surgical excision is required for the management of mature lesions.
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Atypical hemolytic uremic syndrome (aHUS) is a rare and complex disease characterized by microangiopathic hemolytic anemia, thrombocytopenia, and acute renal failure. This case report details the clinical presentation, diagnosis, and management of a 49-year-old female who developed aHUS following elective hip arthroplasty. The patient, with a history of cardiovascular events and no prior renal disease, presented with elevated LDH levels, thrombocytopenia, and acute renal failure on the first postoperative day. A diagnostic workup confirmed aHUS, and the patient was successfully treated with therapeutic plasma exchange (TPE) and hemodialysis. The case underscores the importance of early recognition and aggressive management of aHUS, especially in the perioperative setting, and highlights the need for a multidisciplinary approach to optimize patient outcomes. Through this case, we aim to raise awareness about the potential for surgical stress to trigger aHUS and emphasize the critical role of TPE and supportive care in the treatment of this rare condition.
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Soft tissue aneurysmal bone cysts (STABCs) are rare neoplasms histopathologically identical to aneurysmal bone cysts. These benign lesions are characterized by thin, peripheral ossification and no skeletal continuity. STABC may be difficult to distinguish from myositis ossificans (MO) and malignant entities from imaging and fine needle aspiration, due to rarity and overlapping features. We present a case of a STABC occurring in the paraspinal cervical muscles. The imaging, histopathology, molecular analysis, and treatment are discussed. Four other published cases of STABC of the head and neck are reviewed.
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Myositis ossificans (MO) is characterized by benign heterotopic ossificans in soft tissues like muscles, which can be classified into nonhereditary MO and fibrodysplasia ossificans progressiva (FOP). Although MO has been studied for decades, no research reviewed and analyzed the features of publications in this field quantitatively and qualitatively. Using bibliometrics tools (bibliometrix R package, VOSviewer, and CiteSpace), we conducted a bibliometric analysis of 1280 articles regarding MO in the Web of Science Core Collection database from 1993 to 2022. The annual number of publications and related research areas in the MO field increased gradually in the past 20 years. The USA contributed the most percentage (42.58%) of articles. The University of Pennsylvania (UPenn) and the Journal Bone published the most articles among all institutions and journals. Kaplan FS and Shore EM from UPenn were the top two scholars who made the largest contributions to this field. Keyword analysis showed that research hotspots changed from traumatic MO and clinical management of MO to the genetic etiology, pathogenesis, and treatment of FOP. This study can provide new insights into the research trends of MO and helps researchers grasp and determine future study directions more easily.
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BACKGROUND: Myositis ossificans (MO) is a rare disease involving the formation of bone outside the musculoskeletal system. While surgical intervention is the main treatment approach, preventing recurrence and standardized rehabilitation are also crucial. Here, we present a surgical strategy to prevent the recurrence of MO. CASE SUMMARY: A 28-year-old female patient was admitted for the first time for a comminuted fracture of the left olecranon. However, incorrect postoperative rehabilitation resulted in the development of elbow joint stiffness with ectopic ossification, causing a loss of normal range of motion. The patient was diagnosed with MO based on physical examination, X-ray findings, and clinical presentation. We devised a surgical strategy to remove MO, followed by fixation with an Ilizarov frame, and implemented a scientifically reasonable rehabilitation plan. The surgery lasted for 3 h with an estimated blood loss of 45 mL. A drainage tube was placed after surgery, and fluid was aspirated through ultrasound-guided puncture. The patient experienced a significant reduction in joint stiffness after surgery. In the final follow-up at 9 mouths, there was evident improvement in the range of motion of the elbow joint, and no other symptoms were reported. CONCLUSION: The Ilizarov frame is an advantageous surgical technique for facilitating rehabilitation after MO removal. It offers benefits such as passive recovery, individualized treatment, and prompt recovery.
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Myositis ossificans (MO) is a benign condition where bone forms within muscles due to increased activity of the periarticular tissues. Trauma is the most common cause. Nontraumatic MO is exceedingly rare. We present a rare instance of nontraumatic MO affecting the hip in a 32-year-old patient. The patient had a known case of seizure disorder and also had a history of a cerebrovascular accident (CVA). Despite the absence of trauma or known predisposing factors, the patient developed a sizable mass in the left hip, causing pain and restricted range of motion (ROM). Surgical excision of the mass was successful, resulting in complete removal and subsequent improvement in hip function and pain relief during postoperative recovery. Histopathological examination confirmed the diagnosis of MO. The patient's ROM normalized, and there were no signs of recurrence at the one-year follow-up. This case highlights the importance of recognizing MO in hip pain cases without trauma. Timely surgery through the approach described effectively removes the mass, preventing recurrence without compromising vital structures. It showcases a successful multidisciplinary approach for rare musculoskeletal conditions, offering valuable insights into similar cases.
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Positron emission tomography/computed tomography (PET/CT) using 18F-fluorodeoxyglucose ([18F]-FDG) is a widely adopted imaging modality for detecting hypermetabolic lesions. However, emerging positron-emitting tracers, such as radiopharmaceuticals featuring fibroblast activation protein (FAP) inhibitors (FAPI) labeled with [18F] or [68Ga], have opened new avenues in nuclear medicine. This case report focuses on the unique behavior of [68Ga]-FAPI in bilateral gluteal myositis ossificans, an infrequent condition characterized by soft tissue ossification. A 45-year-old woman with gastric adenocarcinoma underwent subtotal gastrectomy and received neoadjuvant and adjuvant chemotherapy; [68Ga]-FAPI PET revealed metastatic processes and unexpected [68Ga]-FAPI avid intramuscular ossifications in the pelvic and bilateral thigh muscles. Even though there was no history of trauma, the patient was diagnosed with myositis ossificans, a condition marked by non-cancerous ectopic ossifications. Diagnosis relies on history, radiology, and/or histology. FAPI imaging, increasingly used for inflammatory and infectious diseases, can exhibit uptake in benign conditions, including those involving bones and joints. This case report is the first to document incidental bilateral [68Ga]-FAPI uptake in bilateral gluteal myositis ossificans. The robust [68Ga]-FAPI activity in myositis ossificans highlights the importance of considering myositis ossificans in the context of soft tissue calcifications with intense [68Ga]-FAPI uptake.
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Myositis ossificans, a benign tumor composed of spindle cells and osteoblasts, can clinically and radiologically mimic osteosarcoma. While recognition and accurate diagnosis of myositis ossificans can be a challenge, this is critical as it may allow a conservative surgical approach to maximize functional outcomes. Herein, we present a patient with surface myositis ossificans confirmed genetically by the presence of COL1A1::USP6 gene fusion, along with a literature review. Due to the enhanced visualization of the bone matrix, computed tomography (CT) imaging may be a superior imaging modality to magnetic resonance (MR) imaging. Staged biopsies with samples obtained from the periphery and center of the lesions may allow pathologists to discern the zonal distribution histologically. Furthermore, immunohistochemistry fluorescence in situ hybridization and molecular testing can aid in the distinction of myositis ossificans from mimics. Because of their resemblance to other bone tumors, these cases of myositis ossificans highlight the importance of a multidisciplinary approach integrating clinical, radiologic, and pathologic analysis and involving serial imaging, sampling, and judicious use of ancillary immunohistochemical and molecular testing.
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Miositis Osificante , Osteosarcoma , Humanos , Neoplasias Óseas/patología , Neoplasias Óseas/diagnóstico , Neoplasias Óseas/diagnóstico por imagen , Colágeno Tipo I/genética , Colágeno Tipo I/análisis , Cadena alfa 1 del Colágeno Tipo I , Diagnóstico Diferencial , Inmunohistoquímica , Hibridación Fluorescente in Situ , Imagen por Resonancia Magnética , Miositis Osificante/diagnóstico , Miositis Osificante/patología , Miositis Osificante/diagnóstico por imagen , Miositis Osificante/genética , Osteosarcoma/diagnóstico , Osteosarcoma/patología , Osteosarcoma/diagnóstico por imagen , Tomografía Computarizada por Rayos X , Ubiquitina TiolesterasaRESUMEN
BACKGROUND: This review aims to study the clinical characteristics, diagnostic results, treatments, and outcomes in patients with heterotopic ossification following COVID-19 infection. METHODS: A literature search for eligible articles was conducted using MEDLINE/Pubmed, Global Health, and Scopus databases (January 12th, 2023), including all case reports and case series from any country and language. The criteria for inclusion in this review were cases of COVID-19 infection subsequently developing heterotopic ossification. RESULTS: This systematic review analysed 15 reports (n = 20 patients) documenting cases of heterotopic ossification following COVID-19 infection. 80% of the patients were male, with a median age of 59 years. All patients required intensive care unit stay with an average duration of 48.5 days. Mechanical ventilation was necessary for all patients and 30% of them underwent tracheostomy. Common symptoms included stiffness and pain, most frequently affecting multiple locations (70%), with the hips and shoulders being predominantly involved. X-rays were the most commonly used imaging modality, followed by computed tomography. Although treatment was given, some of the patients continued to experience symptoms, particularly stiffness. CONCLUSION: 20 patients who developed heterotopic ossification after COVID-19 have been reported, the majority of which had at least two independent risk factors for this condition. The link between those two clinical entities is therefore uncertain, requiring further investigation. It is nonetheless important to suspect heterotopic ossification in patients with severe COVID-19 infection, prolonged immobilisation, mechanical ventilation, who develop joint pain and stiffness, as this condition can significantly impact patients' quality of life. PROTOCOL REGISTRATION: CRD42023393516.
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COVID-19 , Osificación Heterotópica , Humanos , Osificación Heterotópica/etiología , Osificación Heterotópica/diagnóstico por imagen , COVID-19/complicaciones , Masculino , Persona de Mediana Edad , Femenino , Respiración Artificial , Anciano , SARS-CoV-2 , AdultoRESUMEN
Heterotopic ossification is the inappropriate formation of bone in soft tissues of the body. It can manifest spontaneously in rare genetic conditions or as a response to injury, known as acquired heterotopic ossification. There are several experimental models for studying acquired heterotopic ossification from different sources of damage. However, their tenuous mechanistic relevance to the human condition, invasive and laborious nature and/or lack of amenability to chemical and genetic screens, limit their utility. To address these limitations, we developed a simple zebrafish injury model that manifests heterotopic ossification with high penetrance in response to clinically emulating injuries, as observed in human myositis ossificans traumatica. Using this model, we defined the transcriptional response to trauma, identifying differentially regulated genes. Mutant analyses revealed that an increase in the activity of the potassium channel Kcnk5b potentiates injury response, whereas loss of function of the interleukin 11 receptor paralogue (Il11ra) resulted in a drastically reduced ossification response. Based on these findings, we postulate that enhanced ionic signalling, specifically through Kcnk5b, regulates the intensity of the skeletogenic injury response, which, in part, requires immune response regulated by Il11ra.
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Osificación Heterotópica , Proteínas de Pez Cebra , Pez Cebra , Animales , Pez Cebra/genética , Osificación Heterotópica/genética , Osificación Heterotópica/patología , Proteínas de Pez Cebra/genética , Proteínas de Pez Cebra/metabolismo , Regulación de la Expresión Génica , Envejecimiento/genética , Envejecimiento/patología , Heridas y Lesiones/complicaciones , Heridas y Lesiones/genética , Heridas y Lesiones/patología , Modelos Animales de Enfermedad , Mutación/genéticaRESUMEN
Myositis ossificans is uncommon in patients with nontraumatic brain injuries. This report presents a challenging case in which myositis ossificans was diagnosed and treated by medical management in a patient who was unable to complain of any symptoms due to akinetic mutism that occurred after nontraumatic subarachnoid hemorrhage. The patient had intermittent high-grade fever, and laboratory tests showed elevated C-reactive protein and D-dimer levels without clinical signs of infection two months after subarachnoid hemorrhage. Lower-extremity venography using computed tomography was performed to rule out deep venous thrombosis. There was no thrombus, but right vastus medialis muscle showed inflammatory change with faint multilayered curvilinear hyperdense rims. The administration of indomethacin helped prevent abnormal bone formation. For the early detection of myositis ossificans, careful observation of clinical presentation and a high index of clinical suspicion is necessary in brain-injured patients. Further, elevated serum inflammatory markers accompanied by elevated alkaline phosphatase can be a critical clue. Early computed tomography helps identify early 'string sign' prior to characteristic ossification. Our report highlights that the myositis ossificans is remediable by early detection and appropriate nonsurgical management.
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A woman in her 20s with a past medical history of surgical debulking of a right neck mass presented to the hospital for persistent and worsening right shoulder pain. The shoulder pain was associated with trismus and back and neck pain. A CT scan of the neck with contrast revealed post-surgical changes with increased heterotopic ossification throughout the surgical site extending to the supraclavicular soft tissues and the left sternocleidomastoid muscle, suggesting muscle ossification. A biopsy was performed, and the patient was diagnosed with myositis ossificans (MO). Initial treatment began with the administration of steroids and analgesics. She was scheduled for a follow-up with orthopedics, rheumatology, and genetics, but she was lost for follow-up. MO is a very rare medical condition usually associated with trauma, and in our patient, the symptoms started after a chiropractic adjustment.
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Functional leg length discrepancy results from soft tissue tightness or weakness across any joint in the lower extremity or spine. Herein, we present a 23-year-old female patient with leg length discrepancy due to a nontraumatic myositis ossificans (MO). Interpretation of the imaging findings is quite decisive in diagnosing soft tissue pathologies. It is particularly valid for MO to differentiate from other malignant or infectious lesions. There is no consensus on the treatment of nontraumatic MO. Although there are studies stating the contrary, surgical interventions should be considered as second option for patients who failed with nonsurgical treatments such as physical therapy.
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Myositis ossificans is delineated and distinguished by the generation and deposition of cartilaginous and osseous soft tissues. It generally occurs in the lower extremities and is caused by direct trauma. During the different developmental stages of maturation, the lesion has different radiological appearances that can be confused with sarcomas. Here, we present the case of a 38-year-old woman who presented to the outpatient clinic with a painful mass in the lateral chest wall that had rapidly expanded and increased in size. The patient had no history of trauma. Chest computed tomography revealed an intramuscular mass in the lateral chest wall; postcontrast images demonstrated heterogeneous enhancement and peripheral calcification. The patient was then referred to our center for subsequent assessment and examinations. Pathological examination findings confirmed the diagnosis of myositis ossificans. Surgical resection was performed after obtaining patient consent. The symptoms experienced by the patient were successfully relieved, and no evidence of recurrence was observed during the 2-year follow-up period. Knowledge of the atypical locations of myositis ossificans, calcification patterns at different stages, and radiopathological correlations can help accurately diagnose myositis ossificans and avoid unnecessary medical imaging and interventions.
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Teaching point: Myositis ossificans is a do not touch lesion of which the radiological findings can be misleading in the early stages due to possible features mimicking a malignant process.
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BACKGROUND: Fibrodysplasia ossificans progressiva (FOP) as a rare and heritable disorder with the infrequent genetic transmission of the condition is a catastrophic disorder of heterotopic ossification (HO) and a cause of extraskeletal bone formation in humans. Given the lack of effective treatment for this disease, the important point is to avoid aggravating factors such as bone biopsy, surgery, and intramuscular injection. CASE PRESENTATION: In this report, we present a 52-year-old female patient, Kurdish ethnic, suspected to FOP who had a surgical intervention on the second toe of the right foot, which subsequently, it caused further deterioration of the disease in the person including necrosis and amputation of the distal phalanx of the second toe. CONCLUSIONS: Although, based on our investigation and the available scientific evidence, surgery may a cause for faster progression and worsening of the FOP disorder, but its proof requires further studies.
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Miositis Osificante , Osificación Heterotópica , Femenino , Humanos , Persona de Mediana Edad , Miositis Osificante/diagnóstico , Miositis Osificante/cirugía , Osificación Heterotópica/etiología , Osificación Heterotópica/patología , Dedos del Pie/patología , Huesos/patologíaRESUMEN
Introduction: Bony subungual growths arising on the digital tip are uncommon. Among these, subungual exostosis is a common cause. Case Presentation: We describe a 35-year-old-male patient with a rare occurrence of a fibro-osseus pseudotumor of the digit. It presents with reactive intramembranous ossification, with no continuity with the distal phalanx, helping differentiate it from a subungual exostosis. The patient presented with an asymptomatic subungual growth, lifting up the nail plate distally causing onycholysis. The characteristic clinical, radiological, and histopathological features of the case are described, which helped confirm the diagnosis. Conclusion: Cases with fibro-osseus pseudotumor of the digit reported in literature are reviewed comprehensively. The factors helping differentiate this from the other bony tumors affecting the distal phalanx, including subungual exostosis, myositis ossificans, and osteosarcoma are also highlighted.
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INTRODUCTION: A soft tissue aneurysmal bone cyst is an extremely rare tumor. The objective of the article is to present the clinical, radiological, and histopathological features of a very unusual neoplasm of soft tissues. CASE REPORT: A 13-year-old male patient presented a painful, mobile, and rapidly growing mass on the posteromedial aspect of his left knee. Imaging studies revealed a mass that arose from the medial surface of the distal sartorius muscle, with extension to the subcutaneous fat tissue. It was a well-circumscribed solid tumor with a peripheral rim calcification on plain film, computerized tomography, and ultrasound (zonal phenomenon). On magnetic resonance imaging, a heterogenous mass on T1-weighted images (WI) and T2-WI was seen, with a peripheral hypointense rim in both sequences. An outstanding edema on T2-WI extending to the soft tissue and muscles of the medial compartment of the knee was detected. The mass was resected, and the "tumoral mimickers" histopathological and molecular (next-generation sequencing) diagnoses confirmed a soft tissue aneurysmal bone cyst. A follow-up showed that the patient was free of disease 12 months after surgery. CONCLUSION: Soft tissue aneurysmal bone cyst is a rare tumor. Appropriate clinical and radiological correlation should be performed to differentiate it from other tumor mimickers.