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.

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.

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.

An Unusual Case of Severe Trismus After Craniotomy.

Myositis ossificans Traumatica (MOT) is a non-neoplastic, heterotrophic ossifying disease process in muscle and soft tissue rarely involving the craniofacial area. Treatment depends on the extent of ossification and functional limitations. We present the first case of MOT of the temporalis muscle following neurosurgical intervention. This is the case of a 28-year-old male patient who underwent numerous neurosurgical interventions and reported progressive trismus following these interventions. Imaging demonstrated a right temporal region radio-opacity consistent with ossification of the temporalis muscle. The patient underwent coronoidectomy with excision of the ossified right temporalis muscle and reconstruction with an anterolateral thigh adipofascial vascular free flap. Postoperatively, he maintained intact function with increased incisal opening and no cosmetic defects. Limited mouth opening after the neurological procedure has a wide differential, and MOT must be considered. Surgical repair and reconstruction are multifaceted problems. Free flap reconstruction should be considered in multiple operated patients.

Zygomaticocoronoid ankylosis with possible myositis ossificans: a very rare case.

Ankylosis forming between the zygomatic arch and the coronoid process is a rarely encountered pathological extracapsular ankylosis. Its treatment protocol consists of surgical removal of the coronoid process with the ankylotic mass and jaw opening-closing exercises after surgery. Myositis ossificans (MO) is a self-limiting, benign ossifying lesion. It affects all types of soft tissues including subcutaneous adipose tissue, muscles, tendons and nerves. It is most frequently found in the muscle as a solitary lesion. The clinical appearance of MO is generally in the form of a mass characterized with an ossified soft tissue. When it develops alone, cross-sectional imaging might not be specific, and it may appear similar to worse etiologies. It is suggested multiple imaging modalities should be used in the assessment of a suspicious soft tissue mass. MO is a benign self-limiting disease. In this case report, in the radiographic examination of a 41-year-old female patient, ankylosis between the left coronoid process and the zygomatic bone accompanied by possible MO in the left medial pterygoid muscle was observed. Resection of the coronoid process with the ipsilateral route, resection of the ankylotic mass with the hemicoronal approach and resection of the contralateral coronoid process with the intraoral approach were performed, but the ossified formation in the medial pterygoid muscle was not touched.

Surgical and Radiological Management of Complicated Uterine Leiomyoma Aided by 3D Models in a Patient with Fibrodysplasia Ossificans Progressiva.

BACKGROUND Fibrodysplasia ossificans progressiva (FOP) is a rare autosomal dominant disorder of the connective tissue. Over time, patients with FOP experience decreased range of motion in the joints and the formation of a second skeleton, limiting mobility. Patients with FOP are advised to avoid any unwarranted surgery owing to the risk of a heterotopic ossification flare-up. For patients who do require a surgical procedure, a multidisciplinary team is recommended for comprehensive management of the patient's needs. CASE REPORT A 27-year-old woman with FOP underwent a hysterectomy for removal of a suspected necrotic uterine fibroid. To aid in presurgical planning and management, patient-specific 3-dimensional (3D) models of the patient's tracheobronchial tree, thorax, and lumbosacral spine were printed from the patient's preoperative computed tomography (CT) imaging. The patient required awake nasal fiberoptic intubation for general anesthesia and transversus abdominus plane block for regional anesthesia. Other anesthesia modalities, including spinal epidural, were ruled out after visualizing the patient's anatomy using the 3D model. Postoperatively, the patient was started on a multi-modal analgesic regimen and a course of steroids, and early ambulation was encouraged. CONCLUSIONS Patients with FOP are high-risk surgical patients requiring the care of multiple specialties. Advanced visualization methods, including 3D printing, can be used to better understand their anatomy and locations of heterotopic bone ossification that can affect patient positioning. Our patient successfully underwent supracervical hysterectomy and bilateral salpingectomy with no signs of fever or sepsis at follow-up.

Myositis ossificans causing ulnar neuropathy: a case report.

Myositis ossificans (MO) can compress peripheral nerves and cause neuropathy. We herein describe a patient with ulnar neuropathy caused by MO at the medial elbow. A 28-year-old man with a drowsy mentality and multiple organ damage following a traffic accident was admitted to our hospital. After 3 weeks of postoperative care, the patient's mental status recovered. However, he complained of severe sharp pain in his left medial forearm and fourth and fifth fingers. He exhibited weak fifth finger abduction and wrist adduction. Severe elbow joint pain was elicited during range-of-motion testing of his left elbow. Ultrasound also showed an edematous, enlarged, hypoechoic ulnar nerve lying above the MO, and the MO outwardly displaced the ulnar nerve. Elbow radiographic examination, computed tomography, and magnetic resonance imaging revealed MO development and compression of the left ulnar nerve. The patient underwent surgery; the following day, his left medial forearm pain completely disappeared with slight improvement in the motor weakness of fifth finger abduction. Ultrasound is a useful tool to easily evaluate the presence of MO and compression of peripheral nerves caused by MO.

Heterotopic ossification on the volar surface of the distal radius in a child with fibrodysplasia ossificans progressiva: challenges in surgical excision of a rare condition.

Fibrodysplasia ossificans progressiva (FOP) is one of the genetic and developmental forms of heterotopic ossification. We report a case of FOP on the volar surface of the distal radius, located close to the median nerve and radial artery with neurologic symptoms secondary to median nerve entrapment. The patient underwent surgical excision of the heterotopic lesion followed by radiation therapy. He had no signs of recurrence with more than 1 year of follow-up. Careful microsurgical dissection of the heterotopic mass must be performed to prevent the formation of new painful lesions and iatrogenic neurovascular injury. In this syndrome, the possibility of nerve entrapment due to the heterotopic lesion should be considered as the cause of neuropathic pain. Early genetic testing for confirmation of the suspected diagnosis can avoid having to do an unnecessary biopsy.

Circumscribed Myositis Ossificans of the Masseter Muscle.

ABSTRACT: The authors report a case of myositis ossificans traumatica of the masseter muscles. A 21-year-old man developed masseter myositis after orthognathic surgery to treat facial asymmetry. The myositis may have been triggered by factors such as calcification of a hematoma, periosteum implantation after surgical trauma, and local soft tissue injury. An intraoral incision was made to excise the ectopic bone, resulting in resolution of the patient's symptoms including limited oral opening.

Myositis ossificans of the thigh causing external compression of the superficial femoral artery and vein: A case report.

RATIONALE: Myositis ossificans (MO) is a benign condition characterized by heterotopic bone formation in the skeletal muscle of extremities. Marked variation can occur in the incidence and location of the bone formed as well as resulting complications. Femoral vessel obstruction caused by MO is an extremely rare but disabling complication. Arterial occlusion may aggravate ischemic conditions, resulting in necrosis in the lower extremity. PATIENT CONCERNS: We report a 41-year-old female with progressive pain and swelling of the right thigh region for 1 year. DIAGNOSES: We diagnosed it as obstruction of the superficial femoral artery and vein caused by external compression of the MO between the sartorius and vastus medialis of the thigh. INTERVENTIONS AND OUTCOMES: Adherent tissues and mass were excised with care without damaging the femoral artery or the vein. However, normal morphology did not recover due to loss of elasticity of femoral vessels. Therefore, after resection of the narrowed region of the femoral artery, a femoral-to-femoral graft interposition using the greater saphenous vein was performed. At 12 months after the surgery, vessel reconstruction computed tomography images confirmed normal continuous flow of the femoral artery. LESSONS: Vascular compression and peripheral inflammatory response due to MO can cause loss of normal vascular morphology. Surgical excision of the mass and the involved femoral artery segment followed by femoral arterial reconstruction should be considered for lesions that do not spontaneously regress to prevent functional impairment and secondary complications in extremities.

When Limb Surgery Has Become the Only Life-Saving Therapy in FOP: A Case Report and Systematic Review of the Literature.

Fibrodysplasia ossificans progressiva (FOP) is a rare disease in which heterotopic ossification (HO) is formed in muscles, tendons and ligaments. Traumatic events, including surgery, are discouraged as this is known to trigger a flare-up with risk of subsequent HO. Anesthetic management for patients with FOP is challenging. Cervical spine fusion, ankylosis of the temporomandibular joints, thoracic insufficiency syndrome, restrictive chest wall disease, and sensitivity to oral trauma complicate airway management and anesthesia and pose life-threatening risks. We report a patient with FOP suffering from life-threatening antibiotic resistant bacterial infected ulcers of the right lower leg and foot. The anesthetic, surgical and postoperative challenges and considerations are discussed. In addition, the literature on limb surgeries of FOP patients is systemically reviewed. The 44 year-old female patient was scheduled for a through-knee amputation. Airway and pulmonary evaluation elicited severe abnormalities, rendering standard general anesthesia a rather complication-prone approach in this patient. Thus, regional anesthesia, supplemented with intravenous analgosedation and N2O-inhalation were performed in this case. The surgery itself was securely planned to avoid any unnecessary tissue damage. Postoperatively the patient was closely monitored for FOP activity by ultrasound and [18F]PET/CT-scan. One year after surgery, a non-significant amount of HO had formed at the operated site. The systematic review revealed seventeen articles in which thirty-two limb surgeries in FOP patients were described. HO reoccurrence was described in 90% of the cases. Clinical improvement due to improved mobility of the operated joint was noted in 16% of the cases. It should be noted, though, that follow-up time was limited and no or inadequate imaging modalities were used to follow-up in the majority of these cases. To conclude, if medically urgent, limb surgery in FOP is possible even when general anesthesia is not preferred. The procedure should be well-planned, alternative techniques or procedures should be tested prior to surgery and special attention should be paid to the correct positioning of the patient. According to the literature recurrent HO should be expected after surgery of a limb, even though it was limited in the case described.

Myositis ossificans mimicking sarcoma: a not so rare bioptic diagnostic pitfall.

BACKGROUND: Myositis ossificans (MO) is a heterotopic bone formation in soft tissues, usually caused by traumas or neuropathies. Although the aetiology remains unclear, MO is supposed to be an osteoblast metaplasia with a benign and self-limiting course. Remarkably, at onset MO can be clinically, radiologically and histologically indistinguishable to soft tissue malignancies, especially in cases lacking a history of trauma, leading to misdiagnoses and improper treatments. CASE PRESENTATION: A 13-year-old male was referred to the Oncology Department because of a previous diagnosis of osteogenic sarcoma of his left thigh. The diagnosis was made upon a history of isolated thigh pain in the absence of traumas, the evidence of a contrast-enhanced soft tissue mass on magnetic resonance imaging and the histological findings of atypical nuclei and mitotic figures. The lesion was eventually radiologically unchanged after five cycles of chemotherapy; thus, the child was referred for radical surgery. At admission, endorsing the child well-appearance, together with the evidence of a reduced calcified lesion on a further magnetic resonance, a clinical suspicion of myositis ossificans was raised. Hence, the excisional biopsy confirmed the pathognomonic zonal pattern of myositis ossificans. CONCLUSIONS: This case highlights some frequent diagnostic pitfalls facing myositis ossificans. A lacking history of traumas, along with a too early radiological and histological evaluation can lead to a misdiagnosis of soft tissue malignancies. Even in the absence of a clear history of trauma, a painful soft tissue swelling with a benign clinical course should raise the suspicion of myositis ossificans.

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.

Pseudomalignant Myositis Ossificans of the Neck in a Child: Case Report and Review of the Literature.

BACKGROUND: Myositis ossificans is a benign process of heterotopic bone formation developing in soft tissues that can mimic malignancy. Differential diagnosis can be difficult without a biopsy when it originates in atypical locations. CASE DESCRIPTION: A 5½-year-old boy was admitted for a cervical tumor causing torticollis. The nodular tumor developed at the lateral border of the right C3-4 foramen, had calcification/ossification at its periphery, and was accompanied by a huge edematous reaction of the scalene muscles. The patient underwent an extensive work-up by pediatric oncologists. A biopsy was requested because of high suspicion of malignancy. At surgery, the lesion was benign on frozen sections and was completely resected, allowing the diagnosis of myositis ossificans. The patient made a rapid and complete recovery. CONCLUSIONS: Myositis ossificans circumscripta is rare in children, especially in the neck region. The diagnostic challenge is to differentiate it from bone and soft tissue malignancies. Appropriate management, including surgery if needed, leads to an excellent outcome. Another concern is to exclude fibrodysplasia ossificans progressiva when atraumatic myositis ossificans develops in a young child in the neck or shoulder region.

Aneursymal Bone Cyst: An Uncommon Paraspinal Tumor in Children.

Paraspinal tumors with benign histology in the absence of trauma rarely arise in children. Treatment of such benign tumors, in contrast to malignancies, generally consists of surgical resection of the lesion with confirmation of histology via pathologic evaluation. We present a pediatric case of an atraumatic paraspinal mass with a histologic diagnosis of aneurysmal bone cyst, and USP6 gene rearrangement supporting the histologic diagnosis. The patient underwent gross total resection of the paraspinal lesion with no additional intervention. We highlight the differential diagnosis of paraspinal tumors in children and key features that led to the diagnosis in this patient.

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.

Nontraumatic Myositis Ossificans as an Uncommon Cause of Scoliosis: Case Report and Review of the Literature.

BACKGROUND: A 5-year-old pediatric patient developed scoliosis associated with nontraumatic myositis ossificans in the lumbar region. Although there have been reports in literature of syndromes leading to widespread muscle ossifications resulting in shoulder deformities owing to impaired movement in the facet joints, to the best of our knowledge there has been no report of scoliosis associated with myositis ossificans. CASE DESCRIPTION: The case presented is of a 5-year-old girl who developed scoliosis associated with nontraumatic myositis ossificans in the lumbar region. On the thoracolumbar radiograph, a hyperintense lesion was seen at the right-side L2-L3 level and scoliosis with a Cobb angle of 16.2° to the right side. The decision for surgery was made with the consideration of the existing scoliosis and that the complaints were associated with paravertebral calcified lesions adjacent to the facet joints. After surgery, the scoliosis improved. CONCLUSIONS: Nontraumatic, paravertebral myositis ossificans at an early age is a very rare pathology. Therefore, it must be recognized that spine deformities such as scoliosis and kyphosis can develop in neglected cases of paravertebral myositis ossificans. In addition, there is a high risk of confusion with malignant pathologies, such as osteosarcoma, in this area. Removal of the mass eliminates both the pain of myositis ossificans and prevents the development of scoliosis.

Simulation-Guided Tracheotomy in a Patient With Fibrodysplasia Ossificans Progressiva.

Fibrodysplasia ossificans progressiva (FOP) is a rare disorder that causes heterotopic bone formation leading to chest wall and spinal deformities. This case describes an 11-year-old female with FOP who presented in respiratory failure necessitating two emergent fiberoptic nasotracheal intubations. The patient had severe trismus, rotary flexion of the neck, and distortion of the airway. A three-dimensional printed model based off of a computed tomography reconstruction was created for an in situ simulation before the true procedure. The surgery and trach change were both uneventful. We propose that with careful preoperative planning, tracheotomy can be an appropriate option for FOP patients. Laryngoscope, 129:812-817, 2019.

[Myositis ossificans circumscripta of the hip: about a case]. / Myosite ossifiant circonscrite de la hanche: à propos d'un cas.

Myositis ossificans circumscripta (MOC) is a rare benign condition characterized by heterotopic ossification occurring in the soft tissues. We report the case of a 15-year old patient complaining of mixed hip pain. No trauma has been reported during the interview. The joint had inflammation appearance and a non-mobilizing swelling adhered to the deeper structure with parietal projection over the groin fold. Radiographs of the hip showed a thickening of the soft tissues of the hip and some periarticular calcifications with unclear appearance. Excisional biopsy of the calcifications was decided and performed. Diagnosis was confirmed by histological examination of the surgical specimen. At six months follow-up the patient was very satisfied with his functional results. Returning to school sports was authorized. The patient had successful outcome.