Painful and restricted hip due to myositis ossificans circumscripta of the pectineus muscle after pelvic fracture: A case report.

RATIONALE: Myositis ossificans circumscripta (MOC) is a rare disorder that causes heterotopic bone formation in soft tissues. It usually occurs after trauma and affects large muscles of the extremities. MOC of the pectineus muscle is extremely rare and has not been reported to be treated surgically. PATIENT CONCERNS: A 52-year-old woman presented with left hip pain and dysfunction 4 months after a traffic accident that caused pelvic and humeral fractures and cerebral hemorrhage. DIAGNOSES: Radiological imaging revealed isolated ossification of the left pectineus muscle. The patient was diagnosed with MOC. INTERVENTIONS: The patient underwent surgical resection of the ossified pectineus muscle followed by local radiation and medical therapy. OUTCOMES: At 12 months postoperatively, she was asymptomatic and had normal hip function. No recurrence was observed on radiography. LESSONS: MOC of the pectineus muscle is a rare condition that can cause severe hip dysfunction. Surgical resection combined with radiation and anti-inflammatory drugs can be an effective treatment option for patients who do not respond to conservative management.

A rare femoral heterotopic bone formation in a 14th-19th century female skeleton from Constância (Portugal).

OBJECTIVE: This paper aims to: (1) document a rare femoral heterotopic ossification (HO), and (2) discuss its aetiology and impact on the individual's locomotion and daily living activities. MATERIALS: Adult female skeleton (SG.14-SK.7) from the village of Constância (Portugal), and dated from the 14th-19th centuries CE. METHODS: The biological profile and the macroscopic analysis of the bone changes were assessed using standardized methods. RESULTS: The macroscopic analysis revealed a large bony mass (8 cm length) located immediately inferior to the small trochanter of the right femur. The lesion exhibited a compact, tubular appearance located at the site of attachment of the pectineus muscle. No signs of bone fracture were observed. CONCLUSIONS: The morphology of the SG.14-SK.7 femoral lesion is compatible with a probable case of myositis ossificans traumatica (MOT), secondary to acute trauma of the pectineus muscle. The underlying trauma episode, such as random accidental and/or occupation-related injury, is unknown. However, it is highly possible that this self-limiting condition significantly impaired the individual's daily life and mobility. SIGNIFICANCE: Evidence of severe acute muscle trauma is a rare finding compared with HO secondary to bone trauma and other minor muscle injuries. Moreover, no cases of MOT affecting the pectineus muscle have been reported in the paleopathological literature to date. LIMITATIONS: Although unlikely, a case of neurogenic or burn-related HO cannot be completely disregarded. It was not possible to undertake radiography as part of this study. SUGGESTIONS FOR FURTHER RESEARCH: The use of imaging techniques to complement the paleopathological description is advised.

Myositis Ossificans with Aneurysmal Bone Cystic Changes at the Thoracic Paraspinal Region: A Case Report.

Myositis ossificans (MO) is a benign heterotopic bone formation in muscle or soft tissue. It is a self-limiting disease that is usually initiated by trauma and often occurs in the extremities of the body. Here we report a rare case of traumatic myositis ossificans caused by unusual trauma (extracorporeal shock wave therapy) at thoracic paraspinal muscles. After a needle biopsy, the lesion increased in size, and the patient's symptoms worsened. Malignant soft tissue tumors such as osteosarcoma should be differentiated, so excision of the mass was performed. The final diagnosis was MO with aneurysmal bone cystic change. This case is a very rare form of MO that showed an unusual cause, location, clinical course, and pathologic result on follow-up. This can be an instructive case for radiologists as it is a common disease entity with unusual manifestations.

Fibrodysplasia Ossificans Progressiva: What Have We Achieved and Where Are We Now? Follow-up to the 2015 Lorentz Workshop.

Fibrodysplasia ossificans progressiva (FOP) is an ultra-rare progressive genetic disease effecting one in a million individuals. During their life, patients with FOP progressively develop bone in the soft tissues resulting in increasing immobility and early death. A mutation in the ACVR1 gene was identified as the causative mutation of FOP in 2006. After this, the pathophysiology of FOP has been further elucidated through the efforts of research groups worldwide. In 2015, a workshop was held to gather these groups and discuss the new challenges in FOP research. Here we present an overview and update on these topics.

Are open surgery and total resection good choices for traumatic myositis ossificans in children?

BACKGROUND: Traumatic myositis ossificans is a self-limited, non-neoplastic, ectopic bone formation in the soft tissues caused by trauma. Conservative treatment is the mainstay of management. However, open surgery and complete resection should be considered in patients with limited joint function and constant pain. Herein, we retrospectively analyzed the outcome of open surgical resection in children with myositis ossificans (MO). METHODS: The data of patients diagnosed with MO between February 2010 and May 2019 were retrospectively analyzed. The clinical data included medical history, physical examination, and laboratory, imaging, and pathological findings. All patients underwent an open surgery and total resection. Radiography findings and joint function were evaluated. Patients with developing complications, such as recurrence and assessing joint function, were followed up. In total, 20 patients (sex, 15 male and 5 female patients; age, 8.1 ± 2.5 years) were included. RESULTS: MO was located around the elbow, wrist, proximal humerus, and thigh in 13, one, one, and five patients, respectively. All patients were followed up for > 12 months. Two patients had partial recurrence after surgery. One patient with forearm MO had poor elbow function (Broberg-Morrey score of 70). Nineteen patients had good functional outcomes according to the Broberg-Morrey and Cooney methods. CONCLUSION: Open surgical resection is an effective method for children with MO. The recurrence rate was low when the surgery was performed after the acute stage (1.5 months from disease presentation).

Myositis Ossificans Traumatica, an Unusual Cause of Mandibular Hypomobility.

ABSTRACT: Myositis ossificans traumatica is a rare condition in which causes restriction of mandibular movement. In this entity, temporomandibular joint is depleted of any problems; although all the patients have the history of trauma to the mandible and the face. Myositis ossificans traumatica can involve other parts of the body like femoral region with higher incidence in compare to the maxillofacial area. Blunt trauma to the head and neck causes muscle contusion injury, hematoma formation in the injured muscles or nearby soft tissues. Calcification of hematoma and heterotopic bone formation in the muscle, soft tissue, or near bony structures causes progressive decrease in mouth opening. Surgical removal of calcified body has been the only and one method for treatment of this disorder but rate of recurrence is high in the literature. The authors want to introduce 2 new cases of myositis ossificans traumatica, and then discuss about etiology and different treatment modalities.

Rehabilitation Approach of a Patient with Myositis Ossificans: Non-surgical Management, Hazard of being Unguided Over Exercised.

Myositis ossificans (MO) is a condition where calcification occurs in the soft tissue as well as around the bone following fracture, vigorous exercise or trauma. Although it is a radiological diagnosis, it often leads physician to an incorrect or missed diagnosis as recurrent fracture. Frequently, it follows haemorrhage into the muscle in the tissue space. We report a 45 years old house-wife presented with the complaints of weakness of right side of body and pain with restricted range of motion (ROM) in right lower limb. She was a diagnosed case of recurrent stroke with rheumatic valvular heart disease. After discharge, vigorous physical exercise was done at home by local physiotherapist without appropriate guidance from physiatrist. Gradually pain was so severe that she didn't allow moving her right lower limb. Over the course of time, she became incapacite and bed bound. She denied any positive family history. With the hip and lower limb problems she consulted with orthopedic surgeon and was referred to Physical Medicine and Rehabilitation department of Bangabandhu Sheikh Mujib Medical University (BSMMU), Dhaka, Bangladesh for further management & rehabilitation. This case is presented to focus on hazard of being unguided, over-exercised and non-surgical management approach of this rare condition.

Rare case of a traumatic myositis ossificans in the tibialis anterior muscle.

Athletes involved in contact sports can be diagnosed with traumatic myositis ossificans. Myositis ossificans is characterised by a benign ossifying lesion in soft tissue mass, most commonly preceded by a muscle contusion in the thigh. Despite the fact that it is often a self-limiting disease, treatment modalities are anti-inflammatory drugs, physiotherapy, shockwave therapy, radiation therapy or surgical resection. We report a 22-year-old competitive football player with severe ongoing pain in the lower leg after a direct trauma. An X-ray showed calcification between the tibia and fibula. An additional ultrasound confirmed the diagnosis: myositis ossificans of the tibialis anterior muscle. After treating him with anti-inflammatory drugs and physiotherapy, he was able to return to sports. Myositis ossificans of the lower leg is an uncommon, though if present, troublesome condition, occurring after a contusion.

Surgical excision of post-traumatic myositis ossificans of the adductor longus in a football player.

A football player was diagnosed with myositis ossificans of his right adductor longus muscle after an acute injury. Conservative treatment failed and 1 year after the initial trauma the patient underwent surgical excision of a large ossification. Seven months postoperatively, the patient was fully recovered and returned to his preinjury activity levels. We present our approach to this case and discuss our considerations, referring to background information about this rare disease.

Myositis ossificans traumatica of the piriformis muscle: a rare mature case in an adult African male.

Myositis ossificans traumatica (MOT) is a common form of heterotopic ossification associated to trauma. Rare mature manifestations and topographically atypical presentations of MOT are often misdiagnosed as osteosarcoma. This case study discusses a rare, mature case of MOT of the piriformis muscle, potentially clinically associated with piriformis syndrome. The ossification was observed on a dry sacral bone of an adult skeleton belonging to a South African male during routine inventory of the Raymond A. Dart Collection of Human Skeletons, the University of the Witwatersrand, Johannesburg. The MOT was located on the anterior aspect of the sacrum at a site corresponding to the upper portion of the origin of the muscle and extended laterally towards the greater trochanter, beyond the greater sciatic notch. It was cylindrical in shape and measured approximately 52.70 mm in length and 12.10 mm in diameter. Micro-focus CT revealed an extensive and mature bony development of the piriformis muscle with distinct outer cortical and inner trabecular bone. In addition, the skeleton showed widespread healed skeletal trauma, suggesting a history of trauma. The MOT was completely fused to the sacral bone excluding the possibility of congenital anomalies. Information on the MOT of the piriformis muscle is vital to clinicians and radiographers to aid in successful diagnosis and management of the piriformis syndrome and sciatica in the gluteal region. This case also provides a rare example to biological anthropologists, paleoanthropologists and bioarchaeologists of the representation of pathologies like these on a dry bone sample.

Two cases of myositis ossificans in children, after prolonged immobilization.

Myossitis ossificans (MO) is a benign disorder characterized by heterotopic bone formation in skeletal muscle. It is divided in three types, fibrodysplasia ossificans progressive (FOP), myositis ossificans circumscripta or traumatica (MOT) and myositis ossificans without a history of trauma (non traumatic or pseudomalignant MO). Myositis ossificans is extremely rare in children younger than 10 years. We present the clinical and radiological findings of two 5-year-old children with pseudomalignant MO due to prolonged immobilization. Plain x-ray films and CT scan with their characteristic findings of mature bone in the periphery of the lesion with smooth contour and well separated from the bone, enabled us to diagnose the lesion. To the best of our knowledge, no such cases have been reported in the literature.

Limb specific Acvr1-knockout during embryogenesis in mice exhibits great toe malformation as seen in Fibrodysplasia Ossificans Progressiva (FOP).

PURPOSE: This study analyzes Prx1-specific conditional knockout of Acvr1 aiming to elucidate the endogenous role of Acvr1 during limb formation in early embryonic development. ACVR1 can exhibit activating and inhibiting function in BMP signaling. ACVR1 gain-of-function mutations can cause the rare disease fibrodysplasia ossificans progressiva (FOP), where patients develop ectopic bone replacing soft tissue, tendons and ligaments. METHODS: Whole-mount in situ hybridization and skeletal preparations revealed that following limb-specific conditional knockout of Acvr1, metacarpals and proximal phalanges were shortened and additional cartilage and bone elements were formed. RESULTS: The analysis of a set of marker genes including ligands and receptors of BMP signaling as well as genes involved in patterning and tendon and cartilage formation, revealed temporal disturbances with distinct spatial patterns. The most striking result was that in the absence of Acvr1 in mesoderm precursor cells, first digits were drastically malformed. CONCLUSION: In FOP, malformation of big toes can serve as a first soft marker in diagnostics. The surprising similarities in phenotype between the described conditional knockout of Acvr1 and the FOP mouse model, indicates a natural inhibitory function of ACVR1. This represents a further step towards better understanding the role of Acvr1 and developing treatment options for FOP.

Bilateral overuse myositis ossificans of the triceps: report of an unusual case.

Overuse myositis ossificans is considered an extremely rare diagnosis, with only a few cases reported so far. A case of a 54-year-old firefighter, involved regularly in training exercises, with bilateral myositis ossificans of the triceps, is presented. The patient had a 4 year history of painful masses at the posterior region of both arms. He never experienced any traumatic event, while physical examination revealed firm masses extending longitudinally within the lateral head of the triceps, bilaterally. The patient had painful restricted range of motion at both sides. Plain X-ray views of the humerus showed linear lobulated heterogeneous masses posterolateral. Surgical resection of both masses, which extended intramuscularly through the whole lateral triceps' head, was performed. Histological examination confirmed the diagnosis of myositis ossificans. Myositis ossificans represents a benign, relative rare clinical entity, defined as heterotopic ossification of the soft tissues. Only 5 cases of myositis ossificans due to overuse have been described so far. The present case is unique, since it is the first one describing bilateral appearance of two masses which extended through the whole length of the lateral head of the triceps. A detailed medical history, as well as imaging examination seem to be necessary in order to establish the diagnosis. Treatment should be decided upon the stage of the lesion. For mature lesions surgical treatment is advised.

An Adult Zebrafish Model of Fibrodysplasia Ossificans Progressiva.

Fibrodysplasia ossificans progressiva (FOP) is a rare human skeletal disease caused by constitutively activating mutations in the gene ACVR1, which encodes a type I BMP/TGFß family member receptor. FOP is characterized by progressive heterotopic ossification (HO) of fibrous tissues, including skeletal muscle, tendons, and ligaments, as well as malformation of the big toes, vertebral fusions, and osteochondromas. Surgical interventions in patients often result in enhanced HO, which can exacerbate rather than improve diagnostic outcomes. As a result of these difficulties, a variety of animal models are needed to study human FOP. Here we describe the methods for creating and characterizing zebrafish conditionally expressing Acvr1lQ204D, the first adult zebrafish model for FOP.

Heterotopic Ossification in Mouse Models of Fibrodysplasia Ossificans Progressiva.

Fibrodysplasia ossificans progressiva (FOP), a rare genetic disorder of progressive extra-skeletal ossification, is the most disabling form of heterotopic ossification (HO) in humans. Most people with FOP carry an activating mutation in a BMP type I receptor gene, ACVR1 R206H, that promotes ectopic chondrogenesis and osteogenesis and in turn HO. Advances in elucidating the cellular and molecular events and mechanisms that lead to the ectopic bone formation are being made through the use of genetically engineered mouse models that recapitulate the human disease. We describe methods for inducing heterotopic ossification in a mouse model that conditionally expresses the Acvr1 R206H allele.