-
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 -
International Journal of Ophthalmology 2024To determine the frequency of patients' vision survival and prognostic factors and evaluate clinical features in rhino-orbital mucormycosis.
AIM
To determine the frequency of patients' vision survival and prognostic factors and evaluate clinical features in rhino-orbital mucormycosis.
METHODS
Forty-three eyes of 43 patients followed up with orbital mucormycosis infections were included in the study. Demographic characteristics of the patients, symptoms at admission, ophthalmologic and non-ophthalmologic examination findings, clinical findings during follow-up, medical and surgical procedures, and complications were recorded. Patient survival was determined by assessing the incidence of mortality, and vision survival was defined as achieving a final visual acuity of at least light perception.
RESULTS
Twenty-seven (62.8%) patients were male, and 16 (37.2%) were female. When the underlying disease status of the patients was examined, it was observed that all patients had an underlying disease and diabetes constituted the majority (65.2%). Periorbital swelling (69.8%) and ophthalmoplegia (53.5%) were the most common symptoms and findings at the admission of patients with mucormycosis infection. The disease resulted in death in 22 (51.2%) patients. The presence of fever and shorter duration of antifungal therapy were associated with lower patient survival. Exenteration surgery was not found to be associated with the survival of the patients. Frozen eye, loss of pupillary light reflex, and development of central retinal artery occlusion were associated with lower vision survival.
CONCLUSION
This study presents one of the most extensive patient series in the literature on rhino-orbital mucormycosis. Knowing the patients' symptoms at the time of admission and the clinical findings during the infection process will increase awareness about the disease.
PubMed: 38766351
DOI: 10.18240/ijo.2024.05.17 -
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 -
Indian Journal of Dental Research :... Oct 2023Pituitary microadenomas are said to be common, with an incidence of 27%. Hypothyroidism causing periorbital oedema (PO) remains rare in the literature, but it is still...
INTRODUCTION
Pituitary microadenomas are said to be common, with an incidence of 27%. Hypothyroidism causing periorbital oedema (PO) remains rare in the literature, but it is still within the etiological factors.
PATIENT CONCERNS
This study presents a 53-year-old patient who presented with PO and visited the dental clinic to exclude dental infection.
DIAGNOSIS
Based on the evaluation of proper haematological and radiological investigations, pituitary microadenoma was identified with an abrupt increase in the thyroid stimulating hormone (TSH) level.
FOLLOWUP
The patient was followed with further studies to identify the primary cause of PO. It was concluded that the disruption in the drug regimen caused an increase in the TSH and led to PO rather than microadenoma.
Topics: Humans; Middle Aged; Hypothyroidism; Edema; Male; Pituitary Neoplasms; Adenoma; Orbital Diseases; Thyrotropin
PubMed: 38739832
DOI: 10.4103/ijdr.ijdr_9_22 -
International Journal of Ophthalmology 2024
PubMed: 38721503
DOI: 10.18240/ijo.2024.03.25 -
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 -
International Archives of... Apr 2024Many patients suffered from rhino-orbital-cerebral mucormycosis during the coronavirus disease 2019 (COVID-19) pandemic in India. Diabetes is a known risk factor of...
Many patients suffered from rhino-orbital-cerebral mucormycosis during the coronavirus disease 2019 (COVID-19) pandemic in India. Diabetes is a known risk factor of COVID-19 infection and mucormycosis. The present study was done to describe the clinical spectrum and histopathological findings of mucormycosis in COVID-19 patients and their outcomes. A cross-sectional study was done over a period of two and half months. The biopsy samples or scrapings from sinonasal or periorbital tissue of 38 patients were analyzed. Hematoxylin & Eosin (H&E stain) slides were evaluated along with Grocott-Gomori methenamine-silver and Periodic acid-Schiff stains to highlight the fungal elements. The male to female ratio was 2.5:1, and the mean age of the subjects was 53 years old. A total of 68.4% ( = 26/38) of the patients had diabetes as a comorbidity, 84.2% ( = 32/38) had a history of steroid intake, and 55.3% ( = 21/38) were given supplemental oxygen during their treatment. The common presentations were nasal blockage, discharge, eye pain, headache, and altered mentation. The sites of biopsy were: nasal cavity 76.3% ( = 29/38), periorbital fat/orbit 21.1% ( = 8/38), maxillary sinus 15.8% ( = 6/38) and ethmoid sinus 13.2% ( 5/38). In 76.3% ( = 29/38) cases, broad, irregular, nonseptate, and right-angle branching hyphae were seen on H&E-stained tissue sections. COVID-19 led to various complications in individuals affected by it. Mucormycosis was one such lethal complication. An early diagnosis and prompt treatment is crucial to control the progression of the disease and improve outcomes.
PubMed: 38618587
DOI: 10.1055/s-0043-1776729 -
International Medical Case Reports... 2024Mucormycosis is a potentially fatal condition with a high mortality rate, particularly when there is extra nasal involvement, and it is rare for patients with fungal...
Mucormycosis is a potentially fatal condition with a high mortality rate, particularly when there is extra nasal involvement, and it is rare for patients with fungal brain disease to survive. It mostly affects patients who are metabolically or immunologically compromised, which constitutes one of the three classical stages of the progression of Rhino-Orbito-Cerebral Mucormycosis (ROCM). Stage I: infection of the nasal mucosa and paranasal sinuses; Stage II: orbital involvement; Stage III: cerebral involvement.Here, we report a case of rhino-orbital cerebral mucormycosis in a 14-year-old girl with no known risk factor who presented with periorbital edema, right eye proptosis, fever, and extreme facial pain, which progressively worsened to confusion and left leg weakness in 3 days after admission. The final diagnosis was rhino-orbital-cerebral mucormycosis. The infection was successfully treated using liposomal amphotericin and surgical debridement to remove infected orbital tissue. Mucormycosis is a potentially fatal disease that necessitates prompt diagnosis and treatment. Children are rarely infected with mucormycosis. The majority of studies show that people are typically between 40 and 50 years old. ROCM is typically diagnosed using clinical symptoms and histopathologic evaluation; however, imaging is critical in determining the presence of intracranial lesion. The standard treatment for ROCM is amphotericin B at a recommended dose of 1.0-1.5 mg/kg/day for weeks or months, depending on the clinical response and severity of adverse drug reactions, particularly nephrotoxicity.Rhino-orbital cerebral mucormycosis in a healthy female child is uncommon; early diagnosis and prompt treatment with Amphotericin B should be necessary. Devastating consequences will result from a delayed diagnosis.
PubMed: 38559497
DOI: 10.2147/IMCRJ.S454697 -
Frontiers in Pediatrics 2024Inborn errors of immunity (IEI) can often be misdiagnosed early in life due to their heterogenous clinical presentations. Interleukin-1 receptor-associated kinase 4...
Inborn errors of immunity (IEI) can often be misdiagnosed early in life due to their heterogenous clinical presentations. Interleukin-1 receptor-associated kinase 4 (IRAK-4) deficiency is one of the rare innate immunodeficiency disorders. We present the case of a patient who presented at the age of 15 days with meningitis and septic shock that responded to antibiotics. She was admitted again at the age of 45 days with bacteremia that was associated with increased inflammatory markers. Her third admission was at the age of 2.5 months due to left sided peri-orbital cellulitis that was again associated with elevated inflammatory markers. At 3.5 months, she experienced left orbital cellulitis, which was complicated by extensive sinus involvement, erosion, and abscess formation in the pterygopalatine fossa. Her condition progressed to septic shock and required multiple antibiotics and surgical interventions for drainage and control of the infection source. Both abscess and blood culture were positive for . An IEI was suspected but basic immunology testing was normal. Whole Exome Sequencing was performed and a novel mutation in IRAK4 was detected. In conclusion, we highlight the importance of raising awareness among pediatricians about the potentially lethal IEI and the need to consult specialists when these diseases are suspected. Among them is IRAK-4 deficiency which can be diagnosed by sophisticated functional assays and/or genetic testing.
PubMed: 38487470
DOI: 10.3389/fped.2024.1340367