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Journal of Ayub Medical College,... 2023We present the case of a 30-year-old woman who presented with 8-month history of intermittent fever, joint pains with morning stiffness, recurrent oral ulcers,...
We present the case of a 30-year-old woman who presented with 8-month history of intermittent fever, joint pains with morning stiffness, recurrent oral ulcers, photosensitivity, weight loss and hair fall. For the last 2 months, she had developed a dry cough with progressive shortening of breath. On examination, a cachexic lady with malar hyperpigmentation, alopecia, pallor, nail dystrophy and erythema over her hands and feet were noted. There were multiple punched-out skin ulcers of variable size over legs, arms and abdomen usually round in shape with well-defined even wound margins and scant serous discharge. Musculoskeletal examination revealed synovitis of both elbows and a few metacarpophalangeal and proximal interphalangeal joints. Chest X-ray and HRCT showed bilateral ground-glass opacification. Anti-Nuclear Antibody (ANA) was positive, 1:320, homogenous nuclear pattern. Anti-Ro antibody was highly positive and serum complement (C3, C4) levels were reduced. She was diagnosed with Lupus Vasculitis and started on steroids, mycophenolate mofetil and hydroxychloroquine.
Topics: Humans; Female; Adult; Cellulitis; Impetigo; Pakistan; Eye Diseases; Mycophenolic Acid
PubMed: 38404102
DOI: 10.55519/JAMC-03-11426 -
IDCases 2024Adult-onset immunodeficiency syndrome is characterized by the presence of anti-interferon-gamma (IFN-γ) autoantibody and the distribution of infections. Here, we...
Adult-onset immunodeficiency syndrome is characterized by the presence of anti-interferon-gamma (IFN-γ) autoantibody and the distribution of infections. Here, we describe bacteremia in a Thai woman who also had anti-IFN-γ autoantibody. The patient was also suffering from osteomyelitis and a peri-orbital abscess. Her symptoms were completely eradicated after surgical intervention and the administration of appropriate antibiotics.
PubMed: 38298220
DOI: 10.1016/j.idcr.2024.e01926 -
Cureus Jan 2024A 24-year-old female with a history of intravenous heroin use presented with two weeks of chills, myalgias, and cough and was found to be in acute hypoxemic respiratory...
A 24-year-old female with a history of intravenous heroin use presented with two weeks of chills, myalgias, and cough and was found to be in acute hypoxemic respiratory failure. Subsequent workup revealed the presence of bilateral septic pulmonary emboli and tricuspid valve endocarditis. Several weeks into her hospitalization, she developed periorbital edema and laboratory testing revealed she had developed acute renal failure and nephrotic range proteinuria. A renal biopsy confirmed the diagnosis of IgA-dominant Staphylococcus-associated glomerulonephritis (IgA-SAGN). Early recognition of this newly recognized variant of glomerulonephritis is paramount, as improper treatment may lead to catastrophic consequences.
PubMed: 38380208
DOI: 10.7759/cureus.52680 -
The Journal of International Medical... May 2024Fungal orbital cellulitis is usually seen in immunocompromised individuals, and opportunistic pathogens are the main etiology. We herein report a case of fungal orbital...
Fungal orbital cellulitis is usually seen in immunocompromised individuals, and opportunistic pathogens are the main etiology. We herein report a case of fungal orbital cellulitis due to in a patient with no history of trauma. A 48-year-old man presented to the emergency room of our hospital with a 2-week history of periorbital swelling, conjunctival hyperemia, and chemosis of his right eye. The visual acuity of his right eye was 6/20, and the intraocular pressure was 44 mmHg. The main clinical findings were proptosis of the right ocular globe with conjunctival hyperemia and a palpable infratemporal orbital mass. Laboratory testing failed to detect the presence of a pathogenic infection, and the lesions on computed tomography images resembled those of a malignant tumor of the orbit. The diagnosis was finally confirmed by postoperative pathological examination, and the patient responded favorably to debridement combined with antifungal therapy. Histopathological examination may help to reveal the nature of this disease. Surgical removal of inflammatory lesions can serve as an important diagnostic and treatment method for fungal orbital cellulitis.
Topics: Humans; Male; Middle Aged; Aspergillosis; Immunocompromised Host; Tomography, X-Ray Computed; Antifungal Agents; Orbital Cellulitis; Debridement; Eye Infections, Fungal
PubMed: 38757522
DOI: 10.1177/03000605241239857 -
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 -
Ear, Nose, & Throat Journal Jun 2024Mucormycosis is an opportunistic fungal disease that affects immunocompromised patients. With the advent of SARS-CoV-2, this opportunistic disease has increased.
OBJECTIVE
Mucormycosis is an opportunistic fungal disease that affects immunocompromised patients. With the advent of SARS-CoV-2, this opportunistic disease has increased.
METHODS
A case series of 47 patients with COVID-19 associated mucormycosis have been analyzed. Demographic information, signs, symptoms, laboratory investigations, imaging studies, and their association with ICU admission and 30-day mortality were assessed.
RESULTS
Total number of 47 consecutive rhino-orbital cerebral mucormycosis (ROCM) cases were analyzed. Periorbital swelling was the most common sign among patients. Majority of cases had diabetes. All patients received liposomal Amphotericin B. Debridement was performed for all cases.
CONCLUSIONS
SARS-CoV-2 increases the susceptibility to mucormycosis infection in various ways. Uncontrolled level of HbA1c in all patients, even non-diabetic individuals, indicates hyperglycemia over the past three months. Diabetes, orbital exenteration, ptosis, periorbital swelling, DKA, LOC, brain involvement, and mechanical ventilation all correlated with a higher rate of ICU admission and 30-day mortality. In addition, a higher white blood cell count is related to the higher probability of ICU admission. While considering all of the inflammatory laboratory data and HbA1c could help predict 30-day mortality.
Topics: Humans; Mucormycosis; Male; Female; COVID-19; Middle Aged; Aged; Adult; SARS-CoV-2; Amphotericin B; Orbital Diseases; Debridement; Antifungal Agents; Immunocompromised Host; Opportunistic Infections; Retrospective Studies; Intensive Care Units
PubMed: 36583239
DOI: 10.1177/01455613221143859 -
Clinical Case Reports Jun 2024Immunosuppression from B-acute lymphoblastic leukemia (B-ALL) chemotherapy and a preceding COVID-19 infection may predispose patients to rare complications such as...
Co-occurrence of rhino-orbital mucormycosis and acute lymphoblastic leukemia post-COVID-19 infection in a young adolescent male: A case report from a low middle-income country.
KEY CLINICAL MESSAGE
Immunosuppression from B-acute lymphoblastic leukemia (B-ALL) chemotherapy and a preceding COVID-19 infection may predispose patients to rare complications such as rhino-orbital mucormycosis. Hence, a high index of suspicion should be maintained by physicians (and oncologists) if patients undergoing B-ALL treatment present with orofacial symptoms and ophthalmological manifestations such as peri-orbital swelling, ophthalmoplegia, and loss of vision, suggestive of infection.
ABSTRACT
Mucormycosis is a severe fungal infection that poses significant mortality and morbidity risks, particularly in immunocompromised individuals. We present a rare case of a 16-year-old patient with rhino-orbital mucormycosis following B-acute lymphoblastic leukemia (B-ALL) treatment and concurrent COVID-19 infection. We describe the clinical presentation, diagnosis, treatment, and outcome of this patient, and discuss the possible interactions and implications of these three conditions. A young 16-year-old male patient without significant clinical history was admitted with complaints of low-grade intermittent fever, fatigue, malaise, restlessness, and unexplained weight loss for the past 2 months. A bone marrow biopsy confirmed the diagnosis of B-ALL. Following the diagnosis of B-ALL, the patient underwent initiation of chemotherapy. Following the initial two cycles of chemotherapy, the patient experienced fever and cough and tested positive for COVID-19 infection. Nearly a week later, the patient presented to the chemotherapy emergency department with a clinical picture characterized by a fever up to 39°C associated with left facial swelling, severe headache, purulent rhinorrhea, and foreign body sensation in the ipsilateral nostril. The following day, erythema and left eyelid edema were observed, with ocular opening limitation. The diagnosis was confirmed based on the positive result of polymerase chain reaction for left-sided mucormycosis. Initial administration of liposomal and lipid amphotericin B at 1-1.5 mg/kg/d doses for 4-6 weeks was followed by surgical debridement of necrotic tissue on the left side of the face and nose. Subsequent ophthalmological examinations showed normal conditions of the left eye. The case underscores the importance of heightened clinical suspicion, early diagnosis through imaging and molecular techniques, aggressive multimodal therapy, and close interdisciplinary collaboration for improved outcomes in such rare and challenging clinical scenarios.
PubMed: 38799526
DOI: 10.1002/ccr3.8972 -
Medicine Apr 2024Trigeminal herpes zoster, which comprises 10% to 20% of cases of herpes zoster, often leads to severe pain in the ophthalmic branches. Current treatments, including drug...
INTRODUCTION
Trigeminal herpes zoster, which comprises 10% to 20% of cases of herpes zoster, often leads to severe pain in the ophthalmic branches. Current treatments, including drug therapy and minimally invasive interventions, have limitations; accordingly, there is a need to explore alternative approaches. This study aimed to evaluate the efficacy and safety of computerized tomography (CT)-guided pulsed radiofrequency of the sphenopalatine ganglion in patients with intractable trigeminal herpetic pain.
PATIENT CONCERNS
Three patients with intractable trigeminal ophthalmic zoster neuralgia were studied. All patients complained of bursts of headache, which occurred at least 10 times a day, usually in the periorbital and frontal regions. Conventional treatments, including oral medications and radiofrequency therapy targeting the trigeminal-semilunar ganglion and supraorbital nerve, could not sufficiently provide relief.
DIAGNOSIS
Two patients were diagnosed with herpes zoster in the ocular branch of the trigeminal nerve with conjunctivitis, while one patient was diagnosed with postherpetic neuralgia in the ocular branch of the trigeminal nerve.
INTERVENTIONS
This study employed a novel approach that involved CT-guided radiofrequency regulation of the pterygopalatine fossa sphenopalatine ganglion.
OUTCOMES
In all three patients, pain relief was achieved within 1 to 3 days after treatment. During the follow-up, one patient had pain recurrence; however, its severity was ≈ 40% lower than the pretreatment pain severity. The second patient had sustained and effective pain relief. However, the pain of the third patient worsened again after 2 months. The average follow-up duration was 3 months. None of the enrolled patients showed treatment-related adverse reactions or complications.
CONCLUSION
Our findings indicated that CT-guided radiofrequency regulation of the pterygopalatine fossa sphenopalatine ganglion was a safe and effective intervention for pain in patients with trigeminal ophthalmic zoster neuralgia, suggesting that it may be a therapeutic option if other treatments fail.
Topics: Humans; Herpes Zoster Ophthalmicus; Pulsed Radiofrequency Treatment; Neuralgia; Neuralgia, Postherpetic; Trigeminal Neuralgia; Herpes Zoster; Pain, Intractable; Treatment Outcome
PubMed: 38640323
DOI: 10.1097/MD.0000000000037884 -
Ear, Nose, & Throat Journal Aug 2023Central retinal artery occlusion (CRAO) is an ophthalmic emergency and has poor visual prognosis. It is commonly found in elderly people and very rare in child. We...
Central retinal artery occlusion (CRAO) is an ophthalmic emergency and has poor visual prognosis. It is commonly found in elderly people and very rare in child. We reported an 8-year-old girl who suffered from acute sinusitis, periorbital swelling, and the visual acuity of her right eye was only light perception. She was diagnosed with CRAO, SPOA (subperiosteal orbital abscess), and acute sinusitis. Emergency treatments including surgery, antibiotics, glucocorticoids, intraocular-pressure-lowering drugs, and vasodilators were taken immediately in order to save the eyesight. The visual acuity of the right eye returned to 20/400. Conclusions: Severe intraorbital complications of acute sinusitis can lead to CRAO. Timely drainage, strong antibiotics, and glucocorticoids are the most effective methods for the treatments.
Topics: Humans; Child; Female; Aged; Abscess; Glucocorticoids; Orbital Cellulitis; Sinusitis; Retinal Artery Occlusion; Acute Disease; Anti-Bacterial Agents; Orbital Diseases
PubMed: 33975449
DOI: 10.1177/01455613211016731 -
American Journal of Ophthalmology Case... Jun 2024Two cases are described of necrotizing Sweet syndrome (nSS), a rare variant of acute febrile neutrophilic dermatosis that mimics necrotizing soft tissue infections.
PURPOSE
Two cases are described of necrotizing Sweet syndrome (nSS), a rare variant of acute febrile neutrophilic dermatosis that mimics necrotizing soft tissue infections.
OBSERVATION
A 74-year-old female with myelodysplastic syndrome (MDS) presented with isolated periorbital nSS that closely mimicked necrotizing fasciitis (NF); she displayed pathergy to debridement, was exquisitely responsive to corticosteroids, and underwent successful first-stage reconstruction of the eyelid with full-thickness skin grafting. A second 40-year-old female patient with relapsed acute myelogenous leukemia (AML) presented with multifocal nSS most prominently involving the eyelid. Positive herpes zoster virus (HSV) PCR and bacterial superinfection complicated the diagnosis. She improved with chemotherapy for AML and corticosteroid therapy.
CONCLUSION
nSS is rare and a high level of clinical suspicion as well as an understanding of its distinguishing features is necessary to avoid undue morbidity. Identification of pathergy, histopathology, microbiology, and clinical context are critical to avoid misdiagnosis of infection.
PubMed: 38487334
DOI: 10.1016/j.ajoc.2024.102033