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Journal of Investigative Medicine High... 2024Dermatomyositis (DM) presents with inflammatory myopathy and distinct skin manifestations, often linked to specific autoantibodies. Anti-transcriptional intermediary...
Dermatomyositis (DM) presents with inflammatory myopathy and distinct skin manifestations, often linked to specific autoantibodies. Anti-transcriptional intermediary factor-1 gamma (TIF-1γ) antibodies (Abs) are typically linked to DM in older patients and malignancy in 15% to 40% of cases. We highlight a case of a 24-year-old female who presented with weakness of proximal muscles, periorbital edema, heliotrope rash, erosions on oral mucosa, and painful scaly rash on the lower extremities. Transcriptional intermediary factor-1 gamma Abs were positive, confirming inflammatory myopathy. Treatment with steroid pulse therapy and immunoglobulin led to improvement. Evaluation for malignancy yielded unremarkable results. This case underscores the importance of recognizing and managing DM with TIF-1γ Ab positive, even in atypical demographics, and highlights the need for comprehensive malignancy evaluation.
Topics: Humans; Female; Dermatomyositis; Autoantibodies; Young Adult; Transcription Factors
PubMed: 38904327
DOI: 10.1177/23247096241263065 -
Pediatrics and Neonatology Jun 2024To evaluate the impact of the pneumococcal conjugate vaccines (PCVs) introduction on the orbital complications of acute rhino-sinusitis (OC-ARS).
OBJECTIVES
To evaluate the impact of the pneumococcal conjugate vaccines (PCVs) introduction on the orbital complications of acute rhino-sinusitis (OC-ARS).
METHODS
A retrospective cohort study of all pediatric patients with OC-ARS during the period 2002-2019. Data included clinical, demographic, laboratory, and microbiology findings. Patients were divided into three groups: before PCV7 introduction (group 1), after PCV7 and before PCV13 (group 2), and after PCV13 (group 3).
RESULTS
Of 265 enrolled patients, 117, 39, and 109 were assigned to groups 1, 2, and 3. During the study period, a significant decrease was recorded in the percentages of patients in Chandler classification severity category 1, with an increase in patients in category 3 (P = 0.011). The yearly incidence of OC-ARS decreased from 12.64 cases per 100,000 population in 2002 to 5.56 per 100,000 in 2008, and 2.99 per 100,000 in 2019 (P < 0.001). Patients aged 0-4 years showed a dramatic decrease from 29 cases per 100,000 population in 2002 to 4.27 per 100,000 in 2019 (P < 0.001). The pathogens retrieved from all cultures performed were Streptococcus pneumoniae (32.5%), non-typeable Haemophilus influenzae (27.5%), Streptococcus Species, (12.5%), and Staphylococcus aureus (20%), with no changes in distribution during the study periods. Surgery was performed in 28 (10.6%) patients.
CONCLUSIONS
A significant decrease was seen in the overall incidence of OC-ARS, mainly attributable to the decrease in patients aged 0-4 years. An increase was recorded in the severity of the disease following PCVs introduction.
PubMed: 38886146
DOI: 10.1016/j.pedneo.2023.12.009 -
Indian Journal of Otolaryngology and... Jun 2024Ophthalmic vein thrombosis is a severe clinical entity with proptosis, eyelid swelling, orbital pain and reduction of visual acuity; its incidence is rare with 3-4 cases...
Ophthalmic vein thrombosis is a severe clinical entity with proptosis, eyelid swelling, orbital pain and reduction of visual acuity; its incidence is rare with 3-4 cases /million /year. Clinical manifestations result from venous congestion caused by septic (orbital cellulitis) or aseptic aetiologies (coagulopathies, trauma) and in some cases it could be associated with cavernous sinus thrombosis. In this paper, we describe a case report unique in the literature, of bilateral cavernous sinus and ophthalmic veins thrombosis due to both septic and aseptic causes characterized by unilateral sphenoid sinusitis sustained by infection. is an opportunistic animal pathogen, and its infections occur in both domestic and wild animals worldwide but are rare in humans; this is the first instance of human infection in the head and neck with an unknown hypercoagulable state.
PubMed: 38883471
DOI: 10.1007/s12070-024-04505-1 -
The American Journal of Case Reports Jun 2024BACKGROUND Morbihan disease, also known as Morbihan syndrome, is a rare medical condition characterized by chronic facial edema predominantly affecting the upper...
BACKGROUND Morbihan disease, also known as Morbihan syndrome, is a rare medical condition characterized by chronic facial edema predominantly affecting the upper two-thirds of the face. Despite being recognized in medical literature for decades, its true prevalence and underlying pathophysiology remain poorly understood. Various hypotheses, including impaired lymphatic drainage, abnormal vascular permeability, immune dysregulation, and inflammatory reactions to demodex infestation, have been proposed to explain the etiology. CASE REPORT We present a case of a 61-year-old man with organized periocular edema of the upper third of the face, ultimately leading to Morbihan disease diagnosis. The patient underwent a midface lift, allowing for tissue retrieval for histopathological examination of the eyelid edematous skin, which revealed chronic inflammation, ectasia of small lymphatic vessels, and features of demodex intrafollicular localization. These findings were not specific, but consistent with the diagnostic hypothesis. The patient was referred to a rheumatologist for further evaluation and treatment. He did not respond well to systemic corticosteroids and immunosuppressive therapy. Rather, this resulted in extension of the edema to the upper eyelid. The patient opted not to undergo further treatment. CONCLUSIONS Morbihan disease is often misdiagnosed due to its rarity and overlapping clinical features with other facial conditions. Its management is challenging and can require a combination of medical and surgical interventions. Systemic corticosteroids, immunosuppressive agents, and topical treatments have had varying success. Surgical procedures, such as blepharoplasty or laser therapy, can be considered in severe cases. Early recognition and appropriate management are crucial to improving patient outcomes and quality of life.
Topics: Humans; Male; Middle Aged; Edema; Mite Infestations; Eyelid Diseases; Animals; Inflammation; Chronic Disease
PubMed: 38872283
DOI: 10.12659/AJCR.943421 -
Indian Journal of Public Health Jan 2024Invasive fungal sinusitis is a highly lethal infection in an immunocompromised population that can spread rapidly to involve the adjacent structures by direct invasion...
Invasive fungal sinusitis is a highly lethal infection in an immunocompromised population that can spread rapidly to involve the adjacent structures by direct invasion or through vascular invasion. Involvement of cerebral parenchyma by vascular invasion is a devastating complication in these patients which may lead to vasculitis, thrombus formation, cerebritis, or abscess formation. Here, we present a case of a young male with uncontrolled diabetes mellitus who initially presented with COVID-19 lung disease and later developed sinonasal mucormycosis complicated with left orbital cellulitis and pulmonary mucormycosis.
Topics: Humans; Mucormycosis; Male; COVID-19; SARS-CoV-2; Lung Diseases, Fungal; Adult; Diabetes Complications
PubMed: 38847641
DOI: 10.4103/ijph.ijph_237_23 -
Ophthalmology Science 2024To describe a pilot study on the use of single-session, high-dose-rate, Food and Drug Administration-cleared, yttrium-90 (Y) plaque brachytherapy for iris and...
PURPOSE
To describe a pilot study on the use of single-session, high-dose-rate, Food and Drug Administration-cleared, yttrium-90 (Y) plaque brachytherapy for iris and iridociliary melanoma.
DESIGN
A single-center, clinical case series.
PARTICIPANTS
Six consecutive patients were included in this study. Each was diagnosed with an iris or iridociliary melanoma based on clinical examination with or without biopsy.
METHODS
Each tumor was staged according to the American Joint Committee on Cancer criteria and received Y eye plaque brachytherapy. The main variables were tumor size, patient age, sex, and method of diagnosis (clinical or biopsy). Surgical techniques, treatment durations, and ocular side effects were recorded. Local control was defined as a lack of tumor growth or regression determined by clinical examinations, including slit-lamp and gonio photography, as well as high-frequency ultrasound measurements. Toxicity parameters included acute and short-term corneal/scleral change, anterior segment inflammation, and cataract progression.
MAIN OUTCOME MEASURES
Local and systemic cancer control, tumor regression, visual acuity, as well as radiation-related normal tissue toxicity.
RESULTS
High-dose-rate Y plaque brachytherapy was used to treat small (American Joint Committee on Cancer cT1) category melanomas. Single-surgery high-dose-rate irradiations were performed under anesthesia. Because of short treatment durations, high-dose-rate Y did not require the additional procedures used for low-dose-rate plaque (e.g., sutures, amniotic membrane epicorneal buffering, Gunderson flaps, and second surgeries for plaque removal). Only conjunctival recession was used to avoid normal tissue irradiation. High-dose-rate Y treatment durations averaged 8.8 minutes (median, 7.9; range, 5.8-12.9). High-dose-rate Y brachytherapy was associated with no periorbital, corneal (Descemet folds), or conjunctival edema. There was no acute or short-term anterior uveitis, secondary cataract, scleropathy, radiation retinopathy, maculopathy, or optic neuropathy. The follow-up was a mean of 16.0 (range 12-24) months. Evidence of local control included a lack of expansion of tumor borders ( = 6, 100%), darkening with or without atrophy of the tumor surface ( = 5/6, 83%), and a mean 24.5% reduction in ultrasonographically measured tumor thickness. There were no cases of metastatic disease.
CONCLUSIONS
High-dose-rate Y brachytherapy allowed for single-surgery, minimally invasive, outpatient irradiation of iris and iridociliary melanomas.
FINANCIAL DISCLOSURES
Proprietary or commercial disclosure may be found in the Footnotes and Disclosures at the end of this article.
PubMed: 38840779
DOI: 10.1016/j.xops.2024.100513 -
Clinical Medicine Insights. Case Reports 2024Infratemporal fossa (ITF) tumors are rare in children and may present with a variety of symptoms. Teratomas are neoplasms derived from the 3 germ layers and...
Infratemporal fossa (ITF) tumors are rare in children and may present with a variety of symptoms. Teratomas are neoplasms derived from the 3 germ layers and approximately 6% to 10% are within the head and neck. Our study discusses one of the first reported cases of teratoma in the ITF in a pediatric patient. A 3-year-old girl presents with 2 years of recurrent monthly left periorbital swelling accompanied by fevers, skin discoloration, and pain. Prior episodes were treated with antibiotics with incomplete resolution. Imaging revealed a cystic lesion centered in the ITF. She was taken for endoscopic endonasal biopsy of the lesion and had no complications. Pathology revealed a mature teratoma composed primarily of pancreatic tissue. Providers should consider masses such as teratoma in the differential for ITF tumors and periorbital edema unresponsive to typical treatment.
PubMed: 38827640
DOI: 10.1177/11795476241255563 -
Cureus May 2024Periorbital necrotizing fasciitis (NF) is a devastating bacterial infection associated with irreversible inflammatory destruction of soft tissues. Outcomes include...
Periorbital necrotizing fasciitis (NF) is a devastating bacterial infection associated with irreversible inflammatory destruction of soft tissues. Outcomes include disfigurement, vision loss, septic shock, and death within hours to days. We describe two cases of periorbital NF that presented to our unit within a three-month period. We aim to highlight the key clinical features of periorbital NF, demonstrate the rapid progression of the disease, and the need for prompt identification and decisive intervention. Both patients presented with fever and left-sided periorbital swelling and showed rapid progression of swelling and gangrenous changes to the periorbital skin with worsening proptosis. They were treated with broad-spectrum intravenous antibiotics and underwent emergency surgical debridement of necrotic tissue followed by reconstruction. We propose a formal protocol that we recommend to aid the diagnosis and management of periorbital NF in an acute setting.
PubMed: 38826916
DOI: 10.7759/cureus.59501 -
Sisli Etfal Hastanesi Tip Bulteni 2024Anthrax is a rare zoonotic disease in humans caused by Bacillus anthracis. The most common form of this disease is cutaneous anthrax. Rarely, eye involvement may occur....
Anthrax is a rare zoonotic disease in humans caused by Bacillus anthracis. The most common form of this disease is cutaneous anthrax. Rarely, eye involvement may occur. In this case, a nine-year-old male patient with anthrax on his left eyelids is presented. From the patient's history, it was learned that a slight papular reaction occurred on the left side of the eye, then the lesion enlarged within three days, and edema developed around the eye. On the fifth day of the patient's preseptal cellulitis diagnosis, progress in eye lesions and necrosis and eschar formation around the eyes were detected, while Bacillus anthracis polymerase chain reaction (PCR) positivity was detected on the fifth day of the patient's complaints. The patient was treated with ciprofloxacin and clindamycin and a clinical response was achieved. Anthrax should be kept in mind in the differential diagnosis of preseptal and orbital cellulitis, especially in patients who have close contact with animals. If palpebral anthrax is not treated effectively on time, it can leave scars on the eyelids and cause permanent deformities and loss of function. Early diagnosis and initiation of antibiotic therapy significantly reduce the occurrence of complications. In this case report, a pediatric case with eyelid anthrax, which is rarely seen in anthrax disease, is presented.
PubMed: 38808049
DOI: 10.14744/SEMB.2023.51261 -
Cureus Apr 2024Intracranial subdural empyema is a loculated collection of pus in the subdural space between the dura mater and the arachnoid that can be life-threatening. Here, we...
Intracranial subdural empyema is a loculated collection of pus in the subdural space between the dura mater and the arachnoid that can be life-threatening. Here, we present a case of a 22-year-old man hospitalized for management of sepsis due to right orbital cellulitis who experienced sudden-onset right-sided hemiplegia and was found to have a holohemispheric intracranial subdural empyema requiring emergent neurosurgical intervention. Subdural empyemas are commonly caused by maxillofacial infections, including orbital infections. We demonstrate that orbital cellulitis may cause an intracranial subdural empyema that can present with sudden-onset neurological deficits warranting prompt neurosurgical intervention.
PubMed: 38800139
DOI: 10.7759/cureus.59065