Research trends and hotspots of myositis ossificans: a bibliometric analysis from 1993 to 2022.

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.

Ilizarov technique for treating elbow stiffness caused by myositis ossificans: A case report.

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.

Surgical Triumph Over Huge Nontraumatic Myositis Ossificans of the Gluteal Region in an Epileptic Patient With History of Stroke: A Case Report.

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.

Incidental Intense Fibroblast Activation Protein Inhibitor (FAPI) Uptake in Bilateral Gluteal Myositis Ossificans: A Case Report.

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.

Genetic regulation of injury-induced heterotopic ossification in adult zebrafish.

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.

Myositis ossificans mimicking bone surface osteosarcoma: case report with literature review.

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.

Heterotopic ossification following COVID-19 infections: systematic literature review of case reports and case series.

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.

Nontraumatic Myositis Ossificans After Spontaneous Subarachnoid Hemorrhage: A Case Report.

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.

Myositis Ossificans Traumatica of Bilateral Sternocleidomastoid Muscles After Chiropractor Adjustment: A Case Report.

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.

Myositis ossificans in the chest wall: A case report.

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.

An unusual clinical presentation of nontraumatic myositis ossificans: Leg length discrepancy.

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.

Myositis Ossificans: A Mimicker of an Intramuscular Tumour.

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.

Disease aggravation following surgery in a rare patient suspected to Fibrodysplasia (Myositis) ossificans progressiva: a case report.

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.

Fibro-Osseus Pseudotumor of the Digit: A Case Report and Review of Literature.

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.

Soft Tissue Aneurysmal Bone Cyst in the Sartorius Muscle of a 13-Year-Old Boy Mimicking Myositis Ossificans: Case Report.

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.

Head and neck manifestations of fibrodysplasia ossificans progressiva: Clinical and imaging findings in 2 cases.

Fibrodysplasia ossificans progressiva is a rare hereditary disorder characterized by progressive heterotopic ossification in muscle and connective tissue, with few reported cases affecting the head and neck region. Although plain radiographic findings and computed tomography features have been well documented, limited reports exist on magnetic resonance findings. This report presents 2 cases of fibrodysplasia ossificans progressiva, one with limited mouth opening due to heterotopic ossification of the lateral pterygoid muscle and the other with restricted neck movement due to heterotopic ossification of the platysma muscle. Clinical findings of restricted mouth opening or limited neck movement, along with radiological findings of associated heterotopic ossification, should prompt consideration of fibrodysplasia ossificans progressiva in the differential diagnosis. Dentists should be particularly vigilant with patients diagnosed with fibrodysplasia ossificans progressiva to avoid exposure to diagnostic biopsy and invasive dental procedures.

Faces of Fibrodysplasia Ossificans Progressiva: Lessons from a Clinical Masquerader.

OBJECTIVES: To evaluate the natural history and to highlight the possible masqueraders causing diagnostic delay and iatrogenic interventions in Fibrodysplasia Ossificans Progressiva (FOP). METHODS: Patient details with suspected FOP were retrieved from the patient registry from 2012 through 2021. Clinical records, X-rays, clinical photographs, and molecular testing results were captured. Follow-up was recorded where available. RESULTS: A total of 16 patients with a clinical diagnosis of FOP were found. Twelve patients with both clinical and molecular records were included in this study. The median age of onset and diagnosis was 1.5 y and 6.5 y respectively with a median diagnostic delay of 3.5 y. The disease course was progressive in ten patients. Seven out of twelve patients were subjected to invasive procedures due to misdiagnosis, which exacerbated their disease progression. CONCLUSIONS: Clinical suspicion followed by molecular testing is straightforward for a confirmed diagnosis of FOP. It is not only diagnostic, cost-effective, and saves time but also avoids unnecessary interventions in these patients.

[Recommendations for the healthcare of patients with FOP]. / Empfehlungen zur Versorgung von Patient:innen mit FOP.

BACKGROUND: Fibrodysplasia ossificans progressiva (FOP) is a very rare, severe genetic disorder triggered by a gain-of-function mutation in the ACVR1 gene that codes for the type I bone morphogenetic protein (BMP) receptor ACVR1 (activin A receptor-type 1), also known as ALK2 (activin receptor-like kinase-2). It leads to the onset and progression of heterotopic ossification (HO) in soft and connective tissue. HO is often preceded by episodes of soft tissue swelling or flare-ups. Flare-ups, characteristic of FOP, may be induced by trauma, infection, vaccination, or other medications, as well as surgical procedures or may occur spontaneously. As patients age, they develop severe mobility limitations due to progressive HO formation, including immobility, causing a shortened life expectancy. FOP's first characteristic clinical sign is the congenital malformation of one or both big toes with valgus axis deviation, which is present in almost all patients. To confirm the diagnosis, molecular genetic analysis of the ACVR1 gene is possible. AIM OF THE RECOMMENDATIONS: This white paper aims to provide an overview of the necessary prerequisites and conditions for the care of patients with FOP and positively contribute to patients with FOP by improving the overall availability of knowledge. To achieve this, relevant aspects of the care of the very rare disease FOP are presented, from the initial diagnosis to the care in regular care based on the authors' knowledge (German FOP network) and the international FOP Treatment Guidelines. The recommendations presented here are addressed to all actors and decision-makers in the health care system and are also intended to inform patients and the public.

Patient-specific PEEK implant for treatment of temporal myositis ossificans (five years follow-up): A case report.

Myositis ossificans of the temporalis muscle results in a cosmetic problem both before and after treatment because of the preoperative swelling and the postoperative defect respectively. The authors hypothesized that a patient-specific Polyether-ether ketone implant can be appropriate for immediate obliteration and reconstruction of such defect benefiting from the accuracy of CAD/CAM technology and computer-guided maxillofacial surgery. A Forty-year-old male patient with myositis ossificans affecting the left temporalis muscle was treated with a computer-guided surgical approach, a patient-specific implant was fabricated to obliterate the defect and avoid temporal hollowing using PEEK material. The functional and cosmetic results were satisfactory both immediately and at the 5-year follow-up, except that the skin over the implant was noticed to be stretched after 5 years. Hence, it can be concluded that virtual surgical planning and PEEK patient-specific implants are reliable in the immediate reconstruction of post-surgical temporal hollowing.

Myositis Ossificans of the Breast Mimicking Breast Cancer: A Case Report.

Myositis ossificans (MO) is an uncommon tumor characterized by a rapidly growing mass following a history of local trauma. Few cases of MO affecting the breast have been reported, and some were misdiagnosed as primary osteosarcoma of the breast or metaplastic breast carcinoma. The following case report presents a patient with a growing breast lump whose core biopsy result was suspicious for breast cancer. MO was diagnosed after analysis of the mastectomy specimen. This case highlights the importance of MO as a differential diagnosis of a growing soft-tissue mass after trauma to avoid unnecessary overtreatment. Keywords: Myositis Ossificans, Osteosarcoma, Breast Cancer, Mastectomy, Heterotopic Ossification © RSNA, 2023.