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Japanese Clinical Medicine 2019Idiopathic orbital myositis, a rare disease of unknown cause, presents a diagnostic puzzle because the diagnosis is based on the exclusion of other diseases. This study...
PURPOSE
Idiopathic orbital myositis, a rare disease of unknown cause, presents a diagnostic puzzle because the diagnosis is based on the exclusion of other diseases. This study aims at elucidating its long-term outcome to answer a clinical question whether idiopathic orbital myositis would be a distinct clinical entity.
METHODS
Retrospective review was made on 7 consecutive patients (6 men and 1 woman) with the age at the initial visit ranging from 33 to 69 years (mean, 45.8 years) who were diagnosed with idiopathic orbital myositis and followed for 5 years or more (mean, 9.2 years) at a referral-based hospital.
RESULTS
Chief complaint at the initial visit was diplopia in 4 patients, blurred vision in 2 patients, and proptosis in 1 patient. On magnetic resonance imaging, 4 patients showed enlargement of a single extraocular muscle on unilateral side while 3 patients showed enlargement of multiple extraocular muscles on unilateral side or bilateral sides. No patient developed systemic diseases or other orbital lesions in the long-term follow-up. All patients at the last visit were free from symptoms, including diplopia, after tapering of prednisolone at the initial dose of 20 to 60 mg daily.
CONCLUSION
Idiopathic orbital myositis had a favorable long-term outcome with corticosteroid administration and appears to be a distinct clinical entity without systemic involvement.
PubMed: 35582015
DOI: 10.1177/1179670719866525 -
Journal of Global Infectious Diseases 2016To study the clinical profile and outcome of dengue fever in children at a tertiary care hospital in Puducherry.
OBJECTIVE
To study the clinical profile and outcome of dengue fever in children at a tertiary care hospital in Puducherry.
MATERIALS AND METHODS
All children (0-12 years of age) diagnosed and confirmed as dengue fever from August 2012 to January 2015 were reviewed retrospectively from hospital case records as per the revised World Health Organization guidelines for dengue fever. The diagnosis was confirmed by NS1 antigen-based ELISA test or dengue serology for IgM and IgG antibodies, and the data were analyzed using SPSS 16.0 statistical software. After collecting the data, all the variables were summarized by descriptive statistics.
RESULTS
Among the 261 confirmed cases of dengue fever non-severe and severe dengue infection was seen in 60.9% and 39.1%, respectively. The mean age (standard deviation) of the presentation was 6.9 + 3.3 years and male: female ratio was 1.2:1. The most common clinical manifestations were fever (94.6%), conjunctival congestion (89.6%), myalgia (81.9%), coryza (79.7%), headache (75.1%), palmar erythema (62.8%), and retro-orbital pain (51.3%). The common early warning signs at the time of admission were persistent vomiting (75.1%), liver enlargement (59.8%), cold and clammy extremities (45.2%), pain abdomen (31.0%), hypotension (29.5%), restlessness (26.4%), giddiness (23.0%), bleeding (19.9%), and oliguria (18.4%). The common manifestation of severe dengue infection was shock (39.1%), bleeding (19.9%), and multi-organ dysfunction (2.3%). The most common complications were liver dysfunction, acute respiratory distress syndrome, encephalopathy, pleural effusion, ascites, myocarditis, myositis, acute kidney injury, and disseminated intravascular coagulopathy. Platelet count did not always correlate well with the severity of bleeding. There were six deaths (2.3%) and out of them four presented with impaired consciousness (66.6%). The common causes for poor outcome were multiorgan failure, encephalopathy, and fluid refractory shock.
CONCLUSION
There has been a resurgence of dengue fever with a change in the pattern of presentation during the recent epidemics. Clinical vigilance and awareness regarding the changing epidemic pattern and timely detection of cases are vital to reduce mortality and morbidity due to severe dengue infection.
PubMed: 27621562
DOI: 10.4103/0974-777X.188596 -
Pediatric Rheumatology Online Journal Apr 2023Sarcoidosis is characterized by non-caseating epithelioid granulomas in various tissues throughout the body, most commonly the lung. Non-caseating granulomas may be seen...
BACKGROUND
Sarcoidosis is characterized by non-caseating epithelioid granulomas in various tissues throughout the body, most commonly the lung. Non-caseating granulomas may be seen in skeletal muscle, though typically asymptomatic and under-recognized. While rare in children, there is a need to better characterize the disease and its management. Here we present a 12-year-old female with bilateral calf pain who was ultimately found to have sarcoid myositis.
CASE PRESENTATION
A 12-year-old female presented to rheumatology with significantly elevated inflammatory markers and isolated lower leg pain. MRI of the distal lower extremities demonstrated extensive bilateral myositis with active inflammation, atrophy, and to a lesser extent fasciitis. This distribution of myositis in a child garnered a broad differential requiring a systematic evaluation. Ultimately, muscle biopsy revealed non-caseating granulomatous myositis with perivascular inflammation, extensive muscle fibrosis, and fatty replacement of the muscle with a CD4+ T cell predominant, lymphohistiocytic infiltrate consistent with sarcoidosis. Review of histopathology from age 6 of an extraconal mass resected from her right superior rectus muscle further confirmed the diagnosis. She had no other clinical symptoms or findings of sarcoidosis. The patient improved significantly with methotrexate and prednisone, though flared again after self-discontinuation of medications and was subsequently lost to follow-up.
CONCLUSION
This is the second reported case of granulomatous myositis associated with sarcoidosis in a pediatric patient, and the first to present with a chief complaint of leg pain. Increased knowledge of pediatric sarcoid myositis within the medical community will enhance recognition of the disease, improve the evaluation of lower leg myositis, and advance outcomes for this vulnerable population.
Topics: Child; Female; Humans; Biomarkers; Fasciitis; Fibrosis; Granuloma; Lower Extremity; Myositis; Pain; Sarcoidosis
PubMed: 37072782
DOI: 10.1186/s12969-023-00816-9 -
Mediterranean Journal of Rheumatology Mar 2024Various muscles can be involved in idiopathic eosinophilic myositis (IEM), with the ocular muscles being notably affected. Ocular eosinophilic myositis is a rare...
Various muscles can be involved in idiopathic eosinophilic myositis (IEM), with the ocular muscles being notably affected. Ocular eosinophilic myositis is a rare condition that typically affects the rectus muscles. A tissue biopsy stands as the gold standard for diagnosis. Different subtypes exist based on the extent of eosinophilic infiltration. Limited data is available about treatment, although glucocorticoids have shown successful outcomes. We present the case of a 60-year-old man who, a few years after being diagnosed with ocular myasthenia gravis, was diagnosed through a tissue biopsy with ocular eosinophilic myositis. Treatment with oral glucocorticoids significantly improved his symptoms.
PubMed: 38736949
DOI: 10.31138/mjr.150523.eom -
Open Access Macedonian Journal of... Jul 2018Orbital myositis is an idiopathic, inflammatory, non-infectious condition, typically confined to more than one extraocular muscle and usually targeting young females in...
BACKGROUND
Orbital myositis is an idiopathic, inflammatory, non-infectious condition, typically confined to more than one extraocular muscle and usually targeting young females in their third decade.
CASE REPORT
We describe a case of orbital myositis uncommonly afflicting an adult male. He initially presented with a sensation of dizziness when turning his head and mobilising, together with right-sided orbital pain that failed to respond to the vestibular sedative cinnarizine. Failure to resolve and development of diplopia initially prompted a working diagnosis of ocular myasthenia gravis. On further investigation using HESS charting, paresis in the inferior and medial rectus and superior oblique ocular muscles of the right eye were elicited correlating with clinical examination. However, the lack of response to low dose steroids and acetylcholinesterase inhibitors, together with a negative screen for myasthenia antibodies, precluded a diagnosis of ocular myaesthenia gravis from being made. Other investigations included a high creatinine kinase and lactate dehydrogenase which indicated pathology in the extraocular muscles themselves. An MRI scan showed normal extraocular muscle thickness while excluding other orbital pathology. Exclusion of a variety of other conditions subsequently led to a diagnosis of orbital myositis.
CONCLUSION
Response to high dose steroids consolidated this diagnosis with a rapid response ascertained clinically by resolution of pseudo-vertigo and pain, as well as the ophthalmoplegia with follow-up on HESS charting. We aim to raise awareness of this rare condition that carries a good response to steroids.
PubMed: 30087737
DOI: 10.3889/oamjms.2018.192 -
Frontiers in Immunology 2020We present a case of a 37-year-old man with HIV infection who had been on antiretroviral therapy for one year. He was admitted to our hospital with red and swollen eyes,...
We present a case of a 37-year-old man with HIV infection who had been on antiretroviral therapy for one year. He was admitted to our hospital with red and swollen eyes, acute onset progressive exophthalmos, and intermittent diplopia endured for 7 days. His symptoms, exam, and imaging led to a diagnosis of immune reconstitution inflammatory syndrome associated orbital myositis. His symptoms improved considerably after glucocorticoid therapy. Following a reduction in the oral prednisone dose, he re-presented with left ptosis, which rapidly progressed to bilateral ptosis. Diagnostic testing led to the diagnosis of immune mediated myasthenia gravis. Treatment with pyridostigmine bromide, prednisone, and tacrolimus was initiated. One month later, the patient's symptoms improved significantly. There was a probable association between his symptoms and autoimmune immune reconstitution inflammatory syndrome. This report highlights the importance of recognizing autoimmune disorders in human immunodeficiency virus-infected patients undergoing antiretroviral therapy. Orbital myositis and myasthenia gravis in human immunodeficiency virus-infected patients correlate closely with immunity status following a marked increase in CD4 T cell counts.
Topics: Adult; HIV Infections; HIV-1; Humans; Immune Reconstitution Inflammatory Syndrome; Male; Myasthenia Gravis; Orbital Myositis; Prednisolone; Pyridostigmine Bromide; Tacrolimus
PubMed: 33381117
DOI: 10.3389/fimmu.2020.595068 -
Cureus Jan 2021An 18-year-old female presented with left eye periorbital swelling, erythema, and pain for three days. Computed tomographic images showed swelling of the medial rectus...
An 18-year-old female presented with left eye periorbital swelling, erythema, and pain for three days. Computed tomographic images showed swelling of the medial rectus muscle, and she was diagnosed with orbital cellulitis and initiated on empiric antibiotics. Over the next 48 hours, she did not clinically improve, resulting in an MRI and further workup of infectious, oncologic, endocrinologic, and rheumatologic etiologies was unrevealing and ruled-out malignancy, sarcoidosis, Wegner's, and thyroid eye disease. Given the negative workup, the presentation was determined to be consistent with idiopathic orbital inflammation (orbital myositis variant) via a diagnosis of exclusion. Therefore, the patient was empirically treated with intravenous steroids that produced pronounced improvement within 24 hours. The patient was discharged in improved condition with a prednisone taper and rheumatology follow-up. Idiopathic orbital inflammation is a rare diagnosis of exclusion in pediatrics that merits prompt consideration and work-up if treatment for orbital cellulitis does not progress as expected.
PubMed: 33585140
DOI: 10.7759/cureus.12655 -
The Canadian Journal of Infectious... Jan 2001Orbital myositis is a common cause of extraocular muscle enlargement. It is characterized by nonspecific inflammation of one or more extraocular muscles. Although often...
Orbital myositis is a common cause of extraocular muscle enlargement. It is characterized by nonspecific inflammation of one or more extraocular muscles. Although often idiopathic in origin, orbital myositis has been associated with various noninfectious diseases. Several cases have also been reported as occurring after upper respiratory tract infections. The present report describes a case of orbital myositis together with subclinical sinusitis and its rapid resolution after antibiotic treatment. The literature on this clinical entity is also reviewed.
PubMed: 18159317
DOI: 10.1155/2001/391964 -
Journal of Current Ophthalmology Sep 2019To evaluate the clinical characteristics, histopathology, and treatment outcomes in adult and pediatric patients with nonspecific orbital inflammation (NSOI).
PURPOSE
To evaluate the clinical characteristics, histopathology, and treatment outcomes in adult and pediatric patients with nonspecific orbital inflammation (NSOI).
METHODS
This retrospective study evaluates 76 patients with NSOI. The patients were categorized in 9 groups according to the site of involvement and histopathology results. These groups included: anterior involvement, dacryoadenitis, myositis, perineural involvement, acute fat involvement, focal mass, orbital apex involvement, diffuse sclerosing form, and multiple tissue involvement. The course of the disease was categorized as acute, subacute, or chronic. The cases with symptom duration of less than 1 week were classified as acute, 1 week to 1 month as subacute, and more than 1 month as chronic.
RESULTS
36 (47.4%) patients were males. The mean age was 41.68 ± 17.62 (6-75) years. The most common signs and symptoms were periorbital pain, periorbital edema, decreased ocular movements or diplopia, and conjunctival injection. The most common group was dacryoadenitis in 29 (38.1%) cases. The most common form of disease was the acute involvement (50% of patients). Most of the patients were treated by oral corticosteroids. Duration of follow-up was 7.17 ± 6.26 months. Recurrence occurred in 9 (11.8%) of patients during the follow-up period.
CONCLUSIONS
This study presents a new categorization in which multiple tissue involvements were separated. Some of the NSOI features differ between adults and children. In most patients, treatment especially with corticosteroids, resolves the clinical findings.
PubMed: 31528770
DOI: 10.1016/j.joco.2019.03.004 -
European Journal of Case Reports in... 2020Extraintestinal manifestations of Crohn's disease sometimes occur and can present prior to intestinal symptoms. Ocular manifestations of Crohn's disease are considered...
UNLABELLED
Extraintestinal manifestations of Crohn's disease sometimes occur and can present prior to intestinal symptoms. Ocular manifestations of Crohn's disease are considered rare, with orbital myositis an even rarer manifestation with only a handful of cases reported in the literature. We present the case of a young woman who was diagnosed with orbital myositis, which was initially attributed to pseudotumor cerebri after an extensive negative work-up. Months later, the patient presented with haematochezia, and was subsequently diagnosed with Crohn's disease.
LEARNING POINTS
Orbital myositis is a rare manifestation of inflammatory bowel disease (IBD), and even more rarely is the initial presentation before the development of intestinal luminal disease.Orbital myositis is a rare extraintestinal manifestation of Crohn's disease and mimics thyroid ophthalmopathy. When a thyroid function test is negative, it is important to keep IBD in mind.
PubMed: 33313010
DOI: 10.12890/2020_001964