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Sisli Etfal Hastanesi Tip Bulteni 2024Numerous neurological manifestations associated with COVID-19 have been reported. However, abducens nerve palsy (ANP) associated with COVID-19 is very rare and mostly...
Numerous neurological manifestations associated with COVID-19 have been reported. However, abducens nerve palsy (ANP) associated with COVID-19 is very rare and mostly related to accompanying respiratory symptoms. Here we present a 29-year-old woman with unilateral ANP manifesting with diplopia and positive SARS-CoV-2 S antibodies, which were checked later. On admission, she had signs of viral pneumonia in thorax CT without any respiratory symptoms. Her cranial neuroimaging revealed no abnormality. Following treatment with favipiravir 2x1600 mg loading dose and then 2x600mg daily maintenance, dexamethasone 8 mg/day and enoxaparin 6000 IU/day, her CT findings recovered completely whereas her ANP only partially resolved. One week after the end of COVID-19 treatment, she also developed Herpes simplex keratitis which was successfully treated with valacyclovir. It should be kept in mind that isolated abducens nerve palsy may be the only finding of COVID-19 cases without any respiratory symptoms.
PubMed: 38808042
DOI: 10.14744/SEMB.2023.55491 -
Cureus Apr 2024Giant cell tumor (GCT) of the skull is an extremely rare condition, accounting for less than one percent of all bone GCTs. Clival GCT is even rarer, with only 25 cases...
Giant cell tumor (GCT) of the skull is an extremely rare condition, accounting for less than one percent of all bone GCTs. Clival GCT is even rarer, with only 25 cases documented to date. It generally follows a benign course; however, due to its location and vascularity, it can be locally aggressive. Complete resection of GCT in this location may be challenging, resulting in residual tumors. In this paper, we report a case of a 19-year-old male who presented with a chronic headache later accompanied by diplopia and was noted to have a mass spanning the sella and the clivus on cranial imaging. The histopathology report of the excised mass revealed findings compatible with GCT of the bone. Most GCTs remain stable in the first two years after initial treatment. However, four months after its partial excision, the clival GCT continued to progress. The patient underwent adjuvant radiation therapy, yet symptoms persisted. This profile highlights the crucial role of long-term surveillance and prompt adjuvant radiation therapy and chemotherapy.
PubMed: 38803788
DOI: 10.7759/cureus.59117 -
Cureus May 2024Sphenoid sinus hemangiomas are uncommon and pose significant diagnostic challenges due to their rarity and the complex symptoms associated with their critical anatomical...
Sphenoid sinus hemangiomas are uncommon and pose significant diagnostic challenges due to their rarity and the complex symptoms associated with their critical anatomical location. This report discusses a woman in her 40s who presented with worsening headaches, diplopia, and a sensation of pressure behind her eyes. Diagnostic imaging revealed a lobulated mass in the sphenoid sinus extending into the cavernous sinus and sella, initially mimicking an aggressive neoplastic pathology. However, histopathological examination following endovascular embolization and partial surgical resection confirmed the diagnosis of a cavernous hemangioma. This case highlights the importance of considering hemangiomas in the differential diagnosis of sphenoid sinus masses, especially when patients present with atypical symptoms and imaging shows features such as high vascularity and bone remodeling. The findings emphasize the need for careful diagnostic and therapeutic approaches to effectively manage such cases and differentiate them from more aggressive pathologies.
PubMed: 38800783
DOI: 10.7759/cureus.61034 -
Frontiers in Immunology 2024Cerebellar ataxia is an uncommon and atypical manifestation of anti--methyl-D-aspartate receptor (NMDAR) encephalitis, often accompanied by seizures, psychiatric...
Cerebellar ataxia is an uncommon and atypical manifestation of anti--methyl-D-aspartate receptor (NMDAR) encephalitis, often accompanied by seizures, psychiatric symptoms, and cognitive deficits. Previous cases of isolated brainstem-cerebellar symptoms in patients with anti-NMDAR encephalitis have not been documented. This report presents a case of anti-NMDAR encephalitis in which the patient exhibited cerebellar ataxia, nystagmus, diplopia, positive bilateral pathological signs, and hemiparesthesia with no other accompanying symptoms or signs. The presence of positive CSF anti-NMDAR antibodies further supports the diagnosis. Other autoantibodies were excluded through the use of cell-based assays. Immunotherapy was subsequently administered, leading to a gradual recovery of the patient.
Topics: Humans; Anti-N-Methyl-D-Aspartate Receptor Encephalitis; Brain Stem; Autoantibodies; Female; Cerebellar Ataxia; Cerebellum; Receptors, N-Methyl-D-Aspartate; Adult; Immunotherapy; Male; Magnetic Resonance Imaging
PubMed: 38799430
DOI: 10.3389/fimmu.2024.1388667 -
Pharmaceutics May 2024Myasthenia gravis (MG) is a rare chronic autoimmune disease caused by the production of autoantibodies against the postsynaptic membrane receptors present at the... (Review)
Review
Myasthenia gravis (MG) is a rare chronic autoimmune disease caused by the production of autoantibodies against the postsynaptic membrane receptors present at the neuromuscular junction. This condition is characterized by fatigue and muscle weakness, including diplopia, ptosis, and systemic impairment. Emerging evidence suggests that in addition to immune dysregulation, the pathogenesis of MG may involve mitochondrial damage and ferroptosis. Mitochondria are the primary site of energy production, and the reactive oxygen species (ROS) generated due to mitochondrial dysfunction can induce ferroptosis. Nanomedicines have been extensively employed to treat various disorders due to their modifiability and good biocompatibility, but their application in MG management has been rather limited. Nevertheless, nanodrug delivery systems that carry immunomodulatory agents, anti-oxidants, or ferroptosis inhibitors could be effective for the treatment of MG. Therefore, this review focuses on various nanoplatforms aimed at attenuating immune dysregulation, restoring mitochondrial function, and inhibiting ferroptosis that could potentially serve as promising agents for targeted MG therapy.
PubMed: 38794313
DOI: 10.3390/pharmaceutics16050651 -
Medicine May 2024The most common subtype of primary lymphoma of the ocular adnexa is the mucosa-associated lymphoid tissue (MALT) subtype. MALT lymphoma of the lacrimal gland is... (Review)
Review
RATIONALE
The most common subtype of primary lymphoma of the ocular adnexa is the mucosa-associated lymphoid tissue (MALT) subtype. MALT lymphoma of the lacrimal gland is relatively rare among the lacrimal gland tumors, and the early clinical symptoms are atypical, which can easily lead to misdiagnosis and missed diagnosis. Here, we report a case of MALT lymphoma of the lacrimal gland and explore its clinical manifestations, pathological characteristics, management, and pathogenesis, with the aim of helping clinicians gain an in-depth understanding of ocular adnexal MALT lymphoma.
PATIENT CONCERNS
A 60-year-old man presented to our hospital with proptosis and diplopia. The right eye deviated and shifted toward the lower part of the nose.
DIAGNOSIS
Orbital enhanced magnetic resonance imaging suggested a mass with a maximum cross-section of 3.2 × 2.1 cm. T1 weighted image was isointense, and the enhancement was more uniform and obvious.
INTERVENTIONS
The right orbital mass was treated surgically, and the final pathology report was MALT lymphoma. After the pathological report was released, the patient was transferred to the hematology department for further diagnosis and no further treatment was given eventually.
OUTCOMES
Seven months later, the patient did not complain of discomfort. Whole-body positron emission tomography-computed tomography, superficial lymph node examination and orbital magnetic resonance imaging revealed no abnormal changes.
LESSONS
The clinical manifestations of MALT lymphoma are heterogeneous. Imaging examination is important for assessing the size of the tumor and its relationship with adjacent tissues. Postoperative pathological examination may provide further evidence for the evaluation of the patient's surgical efficacy and prognosis. Management of MALT lymphoma of the lacrimal gland requires a multidisciplinary approach involving ophthalmologists, hematologists, and radiotherapists.
Topics: Humans; Lymphoma, B-Cell, Marginal Zone; Middle Aged; Male; Eye Neoplasms; Lacrimal Apparatus; Magnetic Resonance Imaging; Lacrimal Apparatus Diseases
PubMed: 38787969
DOI: 10.1097/MD.0000000000038303 -
ENeurologicalSci Jun 2024Waldenstroms macroglobulinemia (WM) is a low-grade B cell neoplasm. Bing Neel syndrome is a rare manifestation of WM characterized by infiltrative involvement of the...
BACKGROUND AND AIMS
Waldenstroms macroglobulinemia (WM) is a low-grade B cell neoplasm. Bing Neel syndrome is a rare manifestation of WM characterized by infiltrative involvement of the central nervous system.
CASE REPORT
64-year-old man, presented with 4 years history of slowly progressive diplopia and ptosis of eyes. Examination showed left oculomotor (internal and external ophthalmoplegia), with trochlear, abducens, and right partial oculomotor and abducens nerve involvement. Evaluation showed anemia of hemoglobin 10.7 g/dL, raised erythrocyte sedimentation rate of 120 mm/h and plasma albumin:globulin reversal. Serum protein electrophoresis showed a paraprotein peak in the early gamma region with elevated IgM level (3810 mg/dL) and elevated free kappa light chain level (70.1 mg/L). Bone marrow aspiration from posterior iliac crest revealed mature small lymphocytes with positive immunohistochemical markers of CD5, CD10 negativity and MYD88 mutation positivity suggestive of WM. Patient was treated with bendamustine and rituximab regimen, with no neurological improvement at the end of one year.
CONCLUSION
This case expands spectrum of paraproteinemic neuropathy to include cranial nerve palsy. Thus, plasma cell dyscrasias have to be considered in patients with isolated ophthalmoparesis especially in elderly patients, even with other comorbidities such as diabetes mellitus.
PubMed: 38784860
DOI: 10.1016/j.ensci.2024.100505 -
Cureus Apr 2024Cranial nerve palsy is common in pituitary disease and depends on the extension of the lesion into the cavernous sinuses. Bilateral cranial nerve palsy was described...
Cranial nerve palsy is common in pituitary disease and depends on the extension of the lesion into the cavernous sinuses. Bilateral cranial nerve palsy was described in pituitary adenomas with apoplexy and in only one case in hypophysitis. We present a case of a 32-year-old female manifesting with headache, diplopia, bilateral sixth nerve palsy, and hypopituitarism. Magnetic resonance imaging (MRI) revealed symmetric expansion of the pituitary gland, with bilateral cavernous sinus invasion and thickening of the pituitary stalk. Hypophysitis was suspected, and after treatment with IV methylprednisolone boluses, a decrease in the pituitary lesion was observed, with complete remission of sixth nerve palsy in the right eye and partial improvement in the left eye. In this case, we report an infrequent form of presentation of hypophysitis, and highlight that steroids are the first line of treatment.
PubMed: 38784347
DOI: 10.7759/cureus.58850 -
Scientific Reports May 2024This study aimed to evaluate the clinical effects, complications (peri- and postoperative), depth of sedation, recovery times, and changes in anxiety levels in... (Randomized Controlled Trial)
Randomized Controlled Trial
This study aimed to evaluate the clinical effects, complications (peri- and postoperative), depth of sedation, recovery times, and changes in anxiety levels in paediatric dental patients receiving intravenous sedation with propofol and ketamine-propofol mixtures. This prospective clinical study included 69 healthy children (ASA 1) aged 3-7 years. The patients were assigned randomly to propofol group (n = 23), which received propofol; 1:3 ketofol group (n = 23), which received 1:3 ketofol; or 1:4 ketofol group (n = 23), which received 1:4 ketofol. The bispectral index (BIS) and Ramsay Sedation Scale (RSS) score were recorded at intervals of 5 min to measure the depth of sedation, and vital signs were evaluated. Peri- and postoperative complications and recovery times were recorded. Anxiety levels were also evaluated using the Facial Image Scale (FIS) and changes in saliva cortisol levels (SCLs) before and after the intravenous sedation procedure. The Kruskal‒Wallis test and Wilcoxon signed-rank test were used to determine pre- and posttreatment parameters. Dunn's test for post hoc analysis was used to determine the differences among groups. Children's pre- and posttreatment anxiety levels did not differ significantly according to FIS scores, and increases in SCLs were detected in 1:3 ketofol and 1:4 ketofol groups after dental treatment was completed. Compared with those in the other groups, the BIS values of the patients in 1:4 ketofol indicated a slightly lower depth of sedation. The recovery time of the patients in 1:3 ketofol was longer than that of patients in propofol and 1:4 ketofol. The incidence of postoperative complications (agitation, hypersalivation, nausea/vomiting, and diplopia) did not differ among the groups. Ketamine-propofol combinations provided effective sedation similar to that of propofol infusion without any serious complications during dental treatment performed under intravenous sedation. The ketofol infusion increased the anxiety level of paediatric dental patients to a greater extent than the propofol infusion.
Topics: Humans; Ketamine; Propofol; Child; Female; Male; Child, Preschool; Prospective Studies; Hypnotics and Sedatives; Conscious Sedation; Anesthesia Recovery Period; Anxiety
PubMed: 38782977
DOI: 10.1038/s41598-024-61823-8 -
F1000Research 2022Pulmonary alveolar proteinosis is a very rare diffuse lung disease characterized by the accumulation of amorphous and periodic acid Schiff-positive lipoproteinaceous...
Pulmonary alveolar proteinosis is a very rare diffuse lung disease characterized by the accumulation of amorphous and periodic acid Schiff-positive lipoproteinaceous material in the alveolar spaces due to impaired surfactant clearance by alveolar macrophages. Three main types were identified: Autoimmune, secondary and congenital. Pulmonary alveolar proteinosis has been previously reported to be associated with several systemic auto-immune diseases. Accordingly, we present the first case report of pulmonary alveolar proteinosis associated with myasthenia gravis. A 27-year-old female patient, ex-smoker, developed a dyspnea on exertion in 2020. The chest X-ray detected diffuse symmetric alveolar opacities. Pulmonary infection was ruled out, particularly COVID-19 infection. The chest scan revealed the "crazy paving" pattern. The bronchoalveolar lavage showed a rosy liquid with granular acellular eosinophilic material Periodic acid-Schiff positive. According to the lung biopsy results, she was diagnosed with pulmonary alveolar proteinosis. The granulocyte macrophage colony-stimulating factor autoantibodies were negative. Nine months later, she was diagnosed with bulbar seronegative myasthenia gravis, confirmed with the electroneuromyography with repetitive nerve stimulation showing significant amplitude decrement of the trapezius and spinal muscles. She was treated with pyridostigmine, oral corticosteroids and azathioprine. Given the worsening respiratory condition of the patient, a bilateral whole lung lavage was performed with a partial resolution of symptoms. Thus, this previously unreported association was treated successfully with rituximab, including improvement of dyspnea, diplopia and muscle fatigability at six months of follow-up. This case emphasizes on the possible association of auto-immune disease to PAP, which could worsen the disease course, as the specific treatment does not exist yet. Hence, further studies are needed to establish clear-cut guidelines for PAP management, particularly when associated to auto-immune diseases.
Topics: Humans; Pulmonary Alveolar Proteinosis; Female; Adult; Myasthenia Gravis
PubMed: 38779463
DOI: 10.12688/f1000research.127299.2