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.

Intra-abdominal myositis ossificans-a clinically challenging disease:A case report

BACKGROUND Myositis ossificans(MO)is an uncommon disorder characterized by heterotopic ossification within soft tissues.Only a few cases of intra-abdominal MO(IMO)have been described in the literature.Histology could be difficult to understand and a wrong diagnosis could lead to an improper cure.CASE SUMMARY We herein report the case of IMO in a healthy 69-year-old man.The patient presented with an abdominal mass in the left lower quadrant.A computed tomography scan showed an inhomogeneous mass with multiple calcifications.The patient underwent radical excision of the mass.Histopathological findings were compatible with MO.Five months later the patient showed a recurrence causing hemorrhagic shock due to intractable intralesional bleeding.The patients eventually died within three months since recurrence.CONCLUSION The case described could be classified as post-traumatic MO that developed close to the previously fractured iliac bone.The subsequent surgical procedure was ineffective and the disease rapidly recurred.The misleading intraoperative diagnosis led to improper surgical treatment with a dramatic evolution.

Giant nontraumatic myositis ossificans in a child:A case report

BACKGROUND Nontraumatic myositis ossificans is a rare disease whose specific pathogenesis is unclear.Early diagnosis of this disease is very difficult in children because of difficulties in determining medical history and nonspecific early clinical manifestations,which may lead to the failure of timely and effective diagnosis and treatment in some patients.We report the diagnosis and treatment of a child with nontraumatic myositis ossificans and summarize the clinical characteristics and diagnosis and treatment of the disease.CASE SUMMARY An 8-year-old girl first came to our hospital for more than a week with pain in the right lower limb.There was no history of trauma or strenuous activities.On physical examination,no mass on the right thigh was found,and the movement of the right lower extremity was limited.Ultrasonography showed synovitis of the hip,and bed rest was recommended.Three days later,the child’s pain persisted and worsened,accompanied by fever and other discomforts.She came to our hospital again and a mass was found on the right thigh with redness and swelling on the surface.The images showed a soft tissue tumor on the right thigh with calcification.Routine blood tests revealed that the inflammation index was significantly increased.In case of infection,the patient was given antibiotics,and the pain was relieved soon after,without fever.However,the right thigh mass persisted and hardened.The patient underwent incision biopsy more than 1 mo later,and the postoperative pathology showed nontraumatic myositis ossificans.After approximately 9 mo of observation,the tumor still persisted,which affected the life of the child,and then resection was performed.Since follow-up,there has been no recurrence.CONCLUSION Due to the difficulty in discerning a child’s medical history and the diverse early manifestations,it is difficult to diagnose nonossifying muscle disease in children in its early stage.Measures such as timely follow-up and periodic image monitoring are conducive to early diagnosis of the disease.The disease has a certain degree of self-limitation,and it can be observed and treated first.If the tumor persists in the later stage or affects functioning,then surgery is considered.

Non Traumatic Myositis Ossificans Mimicking a Malignant Neoplasm: A Case Report

Myositis ossificans is a benign self limiting condition that usually related to trauma. Despite a clinically and histologically distinct entity, myositis ossificans still causes considerable difficulties in diagnosis. A 33-year-old Moroccan woman presented with a 2-month history of left inguinal inflammatory pain with limping gait, MRI examination suggested a malignant neoplasm such as soft tissue osteosarcoma. A diagnosis of myositis ossificans was made by incisional biopsy. Conservative management with clinical and radiological follow up of 19 months confirm the diagnosis. The symptoms resolved within seven months. Myositis ossificans should be considered by clinicians as a possible diagnosis for a soft tissue lesion.

Nontraumatic Myositis Ossificans in a Muscle of Mastication

Myositis ossificans (MO) is an extraskeletal tumor-like lesion with bone formation, and is relatively rare in the head and neck region. We report herein a clinicopathological analysis of MO associated with a muscle of mastication. The patient was a 70-year-old Japanese woman who presented with a tumorous mass in the right parotid area and no history of trauma. The surgically excised tumorous lesion consisted of proliferating fibroblastic cells with no atypia in the central portion, and a formation of trabecular osteoid and/or bony tissue in the peripheral portion as the so-called “zonal phenomenon”. The final histological diagnosis was MO, and the lesion was located in the posterior belly of the digastric muscle on computer tomography. A review of the literature showed that many cases of MO predominantly affected the medial pterygoid and masseter muscle and showed a history of previous trauma. The present case represents a rare case of nontraumatic MO in the uncommon location of the posterior belly of the digastric muscle.

Giant Myositis Ossificans Circumscribed Post Traumatic of Gluteus and Adductor Muscles: Case Report

Myositis ossificans circumscribed is a bone and cartilage heterotopic non neoplastic proliferation inside the soft tissues. It is a benign focal heterotopic ossification process of soft tissues, and a rare disorder that occurs spontaneously or after local trauma. Clinical and radiographic appearances are quite hustler. A careful histological examination of biopsy straightens diagnosis is necessary. There is no consensus in support (surgical or/and medical). From Benign prognosis, evolution of this pathology is usually favorable. The authors report a case of giant myositis ossificans circumscribed post-traumatic localized on gluteus and adductor muscles of the right hip on a 26-year-old man. Through a review of literature the mechanism, the diagnostic methods and therapeutic will be discussed.

Myositis and rhabdomyolysis in scrub typhus infection: A case report

Rationale:Fever with myositis and rhabdomyolysis is a medical emergency requiring prompt diagnosis and management.Scrub typhus associated myositis with rhabdomyolysis is rare.Patient concerns:A 36-year-old female presented with intermittent fever up to 38.6℃,jaundice and progressive weakness of all four limbs.Diagnosis:Scrub typhus associated myositis and rhabdomyolysis.Intervention:Doxycycline 100 mg twice daily and injection of ceftriaxone 1 gm twice daily along with continuous intravenous fluids.Outcome:Fever resolved with normalization of liver function and recovery of muscle power.Lessons:Presence of myositis and rhabdomyolysis is uncommon in scrub typhus;high clinical suspicion should be kept in patients with atypical manifestations of scrub typhus.

Previous Pulmonary Fibrosis in Dermatomyositis/Polymyositis: A Predictive Factor for Pulmonary and Extra-Pulmonary Tuberculosis

Objective: With scant studies in the literature, little is known about the risk factors for tuberculosis in patients with dermatomyositis/polymyositis. Therefore, the aim of the present study was to analyze the predictive factors for tuberculosis development in dermatomyositis/polymyositis. Methods: This single-center, retrospective, cohort study initially included 290 patients with dermatomyositis/polymyositis, from 2002 to 2016. Tuberculosis (pulmonary and/or extra-pulmonary) was confirmed after dermatomyositis/polymyositis diagnosis in 12 patients (4.1%) (Tuberculosis+ group). For the control group (Tuberculosis−), 24 patients without tuberculosis were arbitrarily selected in the same period and matched for age, ethnicity, gender, age at disease diagnosis, disease duration and type (dermatomyositis or polymyositis). Results: Tuberculosis occurred for a median of 16 months after dermatomyositis/polymyositis diagnosis. Clinical, laboratory and treatment features were similar in Tuberculosis+ and Tuberculosis−groups (P > 0.05). However, previous pulmonary fibrosis in dermatomyositis/polymyositis was more prevalent in the Tuberculosis+ group (41.7 vs. 8.3%;P = 0.029). Moreover, on a multivariate logistic regression model, pulmonary fibrosis was significantly associated with Tuberculosis (Odds ratio: 9.59, 95% confidence interval: 1.17 - 78.82). Tuberculosis affected 3 dermatomyositis cases for every 1 polymyositis case, with predominantly pulmonary followed by extra-pulmonary involvement (pleura, cutaneous, muscular, joint, soft tissue and hematologic). Two or more sites were affected in 41.7% of cases. Conclusions: Previous pulmonary fibrosis in dermatomyositis/polymyositis was a predictive factor associated with tuberculosis development. Further studies are needed to confirm these results.

Tofacitinib as a Treatment for Refractory Dermatomyositis: A Case Report

In very scarce case reports and case series, tofacitinib has been a therapeutic alternative for dermatomyositis. To corroborate the literature, we described a refractory dermatomyositis that had a good outcome with tofacitinib. Case Report: An adult female patient presented with definite dermatomyositis and with refractoriness to high doses of intravenous and oral glucocorticoids, intravenous human immunoglobulin, several immunosuppressive drugs (methotrexate, azathioprine, and leflunomide) and two previous immunobiological drugs (rituximab and abatacept). However, the patient had a good outcome with tofacitinib. Conclusions: Tofacitinib appears to be a promising alternative therapy for refractory dermatomyositis.

Non-traumatic myositis ossificans circumscripta at elbow joint in a 9-year old child

Myositis ossificans circumscripta （MOC） is a kind of self-localized, benign and tumor-like lesions often seen in adults, with approximately 75% of cases caused by trauma. We reported a case of non-traumatic MOC occurred at the elbowjoint in a 9-year old child and it has been excised by surgery. After 18 months follow-up, a favorable outcome has been achieved with the Broberg-Morrey score of 100. We suggest that surgical resection should be done as soon as the diagnosis is confirmed.

Clinical Phenotype of Japanese Patients with Dermatomyositis—Classification Based on Dermatomyositis-Specific Autoantibodies

Objectives: To correlate the precise specificity of autoantibodies in Japanese dermatomyositis (DM) patients with their clinical phenotypes. Methods: Serum samples from 94 adult DM patients (67 with classical DM and 27 with clinically amyopathic dermatomyositis, CADM) were screened for autoantibodies using immunoprecipitation assays. Patients with antibodies against aminoacyl transfer RNA synthetase (ARS), Mi-2 or who had other autoantibodies were assessed for clinical symptoms and laboratory findings. Results: Sera from 27 of 94 DM patients (29%) were found to have anti-ARS antibodies. Nineteen (20%) had anti-CADM-140/MDA5, 5 (5%) had anti-Mi-2, and 8 (6%) had anti-p155/TIF1-γ. Anti-MJ/NXP-2 was not found in our series of adult DM. Seventeen patients with anti-ARS had fever and 22 had arthritis and interstitial lung disease (ILD), compatible with a diagnosis of anti-ARS syndrome. Seventeen of 19 (89%) with anti-CADM-140/MDA5 had ILD, 16 (84%) of whom developed rapidly progressive ILD (RP-ILD). Four of 5 (80%) with anti-Mi-2 had heliotrope rash and/or Gottron’s sign/papules, and 2 (40%) had V-sign and/or shawl-sign rash, whereas no ILD or malignancy was detected. As seen with anti-Mi-2-positive patients, a low frequency of ILD (13%) was found in patients with anti-p155/TIF1-γ but 6 of 8 (75%) had malignancy during their course. The frequency of ILD was significantly higher in patients with anti-ARS or anti-CADM-140/MDA5 compared with anti-Mi-2 or anti-p155/TIF1-γ (81% and 89%, respectively). It should be noted that anti-CADM-140/MDA5-positive patients suffered significantly more RP-ILD compared to patients with anti-ARS (84% vs. 7%, P < 0.0001). On the other hand, anti-p155/TIF1-γ positive patients had a significantly higher rate of malignancy compared with anti-ARS-, anti-CADM-140/MDA5-and anti-Mi-2-positive patients (75% vs. 7%: P = 0.0004, 5%: P = 0.0006, 0%: P = 0.02, respectively). Conclusions: These results indicate that in addition to antibodies previously identified as specific for DM, autoantibodies newly found in these patients are useful for stratifying them into clinical subgroups.

Florid reactive periostitis ossificans of the humerus: Case report and differential diagnosis of periosteal lesions of long bones

A case of florid reactive periostitis ossificans(RPO) arising in a long bone is presented. This is a rare bone proliferation with a pronounced periosteal reaction. Less than 100 cases have been described in the literature with far fewer outside the bones of the hand, feet, fingers, and toes. Although the etiology is unknown, a relationship to preceding trauma is suggested. The imaging and histologic features show an overlap with other bone lesions including bizarre parosteal osteochondromatous proliferation, subungual exostosis, and malignant surface tumors of bone and cartilage which include, periosteal and parosteal osteosarcoma. It is important to recognize the clinical presentation and diagnostic features of RPO as a benign entity so that it is not mistaken for a more aggressive neoplasm. We present a case of a right distal humeral lesion that on histopathological review revealed florid RPO. This diagnosis was not suspected on imaging studies, but was made on open biopsy of the mass. The patient remains disease free, years postoperatively. In addition to presenting this unique case report, we review the pertinent literature, and offer a differential diagnosis and treatment strategy for its management.

Proposed criteria to differentiate heterogeneous eosinophilic gastrointestinal disorders of the esophagus, including eosinophilic esophageal myositis

AIM To define clinical criteria to differentiate eosinophilic gastrointestinal disorder(Eo GD) in the esophagus. METHODS Our criteria were defined based on the analyses of the clinical presentation of eosinophilic esophagitis(Eo E), subepithelial eosinophilic esophagitis(s Eo E) and eosinophilic esophageal myositis(Eo EM), identified by endoscopy, manometry and serum immunoglobulin E levels(s-Ig E), in combination with histological and polymerase chain reaction analyses on esophageal tissue samples.RESULTS In five patients with Eo E, endoscopy revealed longitudinal furrows and white plaques in all, and fixed rings in two. In one patient with s Eo E and four with Eo EM, endoscopy showed luminal compression only. Using manometry, failed peristalsis was observed in patients with Eo E and s Eo E with some variation, while Eo EM was associated with hypercontractile or hypertensive peristalsis, with elevated s-Ig E. Histology revealed the following eosinophils per high-power field values. Eo E = 41.4 ± 7.9 in the epithelium and 2.3 ± 1.5 in the subepithelium; s Eo E = 3 in the epithelium and 35 in the subepithelium(conventional biopsy); Eo EM = none in the epithelium, 10.7 ± 11.7 in the subepithelium(conventional biopsy or endoscopic mucosal resection) and 46.8 ± 16.5 in the muscularis propria(peroral esophageal muscle biopsy). Presence of dilated epithelial intercellular space and downward papillae elongation were specific to Eo E. Eotaxin-3, IL-5 and IL-13 were overexpressed in Eo E.CONCLUSION Based on clinical and histological data, we identified criteria, which differentiated between Eo E, s Eo E and Eo EM, and reflected a different pathogenesis between these esophageal Eo GDs.

Left abdominal wall proliferative myositis resection and patch repair:A case report

BACKGROUND Proliferative myositis is a rare benign tumor that is typically self-limiting and does not become malignant.It can be cured by simple resection without reported recurrence.Due to its rapid growth,hard structure and ill-defined borders,it can however be mistaken for malignant tumors such as sarcomas.CASE SUMMARY We investigate the case of a 64-year-old male with proliferative myositis of the abdominal wall,who was preoperatively administered a needle aspiration biopsy and given a simple excision and patch repair.We then compared it with other similar cases to determine the effectiveness of this treatment method.CONCLUSION Resection with follow-up observation has shown to be an effective treatment method for proliferative myositis.To avoid unnecessarily extended or destructive resection,a thorough and conclusive diagnosis is crucial,which requires adequate imaging and pathological knowledge.

Infective myositis A one-case report

BACKGROUND： Infective myositis is rare. The retrospective report of clinical data and symptoms from one patient with infective myositis will hopefully provide more information for clinicians in the diagnosis of this disease. METHODS： A male patient, 65 years old, was admitted with ＂fever and muscle pain since four days ago, accompanied by inertia of all limbs for one day＂, to the First Department of Neurology, Guangdong Provincial Hospital of Traditional Chinese Medicine on April 25th, 2005. Following admission, a history of diseases was record, and detailed physical and neurological examinations were performed. During the examination, symmetrical myasthenia appeared, tendon reflex disappeared, and creatine kinase levels were increased 500 times higher than normal. The patient was primarily diagnosed with hypokalemic periodic paralysis, myositis, Guillain-Barre syndrome, and upper respiratory infection. Subsequently, the patient was treated with the following： cefuroxime for infection, potassium supplements, breviscapin for promoting blood circulation through the removal of stasis, and ATP/CO-A for myocardial nutrition. Antiviral drugs were not administered. However, laboratory samples were continuously monitored. Creatine kinase levels decreased to normal, and muscle pain was obviously relieved following antibiotics treatment. The results led to a final diagnosis of infective myositis. RESULTS： Four days after treatment （April 29th）, muscular tenderness and throat congestion were obviously improved （＋/-）. The neurological examination showed the patient was conscious, cooperated with treatment, and had normal intellect. No abnormalities of the cranial nerve were observed upon examination. Proximal and distal muscle strength and muscular tensions of the four limbs were all normal. Reflexes of the right biceps brachii muscle and its tendon were decreased, and knee tendon and Achilles tendon reflex were not induced. A Babinski reflex was not detected. The neurological examination presented no abnormalities, and the related creatine kinases were within normal range by re-examination at 2 weeks, and l and 3 months after discharge. CONCLUSION： A diagnosis of infective myositis should be considered for patients with clear prodromes of infection, that include symptoms of periodic paralysis and elevated creatine kinase levels. Treatment with sufficient antibiotics can obtain good results.

Effects of Serum from a Fibrodysplasia Ossificans Progressiva Patient on Osteoblastic Cells

Introduction: Fibrodysplasia ossificans progressiva (FOP) is a rare autosomal dominant disorder of skeletal malformations and progressive extraskeletal ossification. Patients and Methods: In the present study, we present a case of FOP with marked progressive ossification of extraskeletal tissues. We investigated whether soluble factors in serum would affect the osteoblast phenotype by examining the effects of serum from this patient and control subjects on mouse osteoblastic MC3T3-E1 cells. Results: The clinical findings of this patient were compatible with FOP, and direct sequence analysis of genomic DNA demonstrated the presence of a heterozygous 617G> A (R206H) mutation of activin type 1 receptor (ACVR1). Serum from the FOP patient enhanced the level of alkaline phosphatase (ALP) in Western blotting, compared with serum from the control, in MC3T3-E1 cells. Moreover, serum from the FOP patient enhanced the levels of ALP, osteocalcin and bone morphogenetic protein-2 mRNA in these cells. Conclusion: We presented a case of FOP with progressive ossification in extra-skeletal tissues with ACVR1 mutation. The present data suggest that the serum from this patient includes some soluble factors, which might enhance the osteoblast differentiation and BMP-2 expression in mouse osteoblastic cells.

An early complication of Transobturator tape: Non-infective adductor internus myositis

The transobturator tape (TOT) procedure is generally felt to be a safer surgical alternative to the ten sion-free vaginal tape procedure for women with stress urinary incontinence. We report a case of adductor internus myositis not associated with infection following the TOT procedure. To our knowledge this is the first case of this type reported in the literature. A 43 year old lady underwent a straightforward elective TOT procedure. There were no intraoperative complications. Immediately following the procedure she complained of pain in her right thigh. MRI confirmed abnormal oedema within the antero-inferior aspect of the right obturator internus muscle consistent with myositis secondary to tape insertion. The tape was removed the following day in theatre, following which her pain resolved. This case highlights a previously unreported complication as a result of the TOT procedure.

Predictive Models of Clinical Improvement in Rituximab-Treated Myositis Patients Using Clinical Features, Autoantibodies, and Biomarkers

Background: Response to rituximab so far is unpredictable in patients with refractory myositis. Predictive models of clinical improvement are developed using clinical, laboratory, and gene expression/cytokine/chemokine variables in rituximab-treated refractory myositis patients. Methods: We analyzed data for 200 myositis patients (76 with adult polymyositis (PM), 76 with adult dermatomyositis (DM), and 48 with juvenile (DM)) in the rituximab in myositis trial. Clinical improvement is defined as the change from baseline to 24 weeks in Physician Global Visual Analog Scale (VAS). We analyze the association of baseline variables with improvements: demographics, myositis subtype, clinical and laboratory parameters, autoantibody status, and interferon (IFN)- regulated chemokines. Multivariable linear regression models are developed by using stepwise variable selection methods. Results: A “base” multivariable model to predict improvement with clinical and laboratory variablesonly is built with modest predictive ability (adjusted R2 = 0.21). This model includes two significant factors at baseline: Physician Global VAS and Muscle Disease Activity VAS. A “final” multivariable model to predict improvement including non-standard laboratory measures is developed and demonstrated better predictive ability (adjusted R2 = 0.32). This model includes Physician Global VAS, IFN chemokine score and IL-2 levels. The “final” model explained 11% more variability than the “base” model. Conclusions: Changes in disease activity over time following treatment with rituximab in refractory myositis can be predicted. These models can be clinically useful to optimize treatment selection in myositis.

Arachnoiditis Ossificans of Lumbosacral Spine: a Case Report and Literature Review

ARACHNOIDITIS ossificans （AO） is a rare type of chronic arachnoiditis presence of calcification spinal arachnoid. Small characterized by the in, or ossification of the plaques of calcificationof the dura mater are frequently observed in radiography or during surgery, which are often asymptomatic and have no apparent clinical significance. AO and dura calcification should not be confused.

Trismus originating from rare fungal myositis in pterygoid muscles:A case report

BACKGROUND Trismus is a common problem with various causes.Any abnormal conditions of relevant anatomic structures that disturb the free movement of the jaw might provoke trismus.Trismus has a detrimental effect on the quality of life.The outcome of this abnormality is critically dependent on timely diagnosis and treatment,and it is difficult to identify the true origin in some cases.We present a rare case of trismus due to fungal myositis in the pterygoid muscle,excluding any other possible pathogenesis.CASE SUMMARY The patient presented with a 2-mo history of restricted mouth opening.Computed tomography showed obvious enlargement of the left pterygoid muscles.Furthermore,the patient had trismus without obvious predisposing causes.The primary diagnosis was pterygoid myosarcoma.Consequently,lesionectomy of the left pterygoid muscle was performed.Intraoperative frozen biopsy implied the possibility of an uncommon infection.Postoperative pathologic examination confirmed myositis and necrosis in the pterygoid muscle.Fungi were detected in both muscle tissue and surrounding necrotic tissue.The patient recovered well with antifungal therapy and mouth opening exercises.The rarity of fungal myositis may be responsible for the misdiagnosis.Although the origin of pathogenic fungi is still unknown,we believe that both hematogenous spread and local invasion could be the most likely sources.To the best of our knowledge,this is the
rst case in the literature that reported fungal myositis in pterygoid muscles as the only reason that results in trismus.CONCLUSION Surgeons should remain vigilant to the possibility of trismus originating from fungal myositis.