Treatment of paediatric vancomycin intoxication: a case report and review of the literature.
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ABSTRACT:
SUBMITTER: Lemaire M
PROVIDER: S-EPMC5477951 | biostudies-literature | 2010 Jun
REPOSITORIES: biostudies-literature
Publications
Treatment of paediatric vancomycin intoxication: a case report and review of the literature.
Lemaire Mathieu M Connolly Bairbre B Harvey Elizabeth E Licht Christoph C
NDT plus 20100317 3
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Project description:Bullous pemphigoid (BP) is a rare, life-threatening autoimmune blistering disease with pruritus and tension blisters/bullous as the main clinical manifestations. Glucocorticosteroids are the main therapeutic agents for it, but their efficacy is poor in some patients. Tofacitinib, a small molecule agent that inhibits JAK1/3, has shown incredible efficacy in a wide range of autoimmune diseases and maybe a new valuable treatment option for refractory BP. To report a case of refractory BP successfully treated with tofacitinib, then explore the underlying mechanism behind the treatment, and finally review similarities to other cases reported in the literature. Case report and literature review of published cases of successful BP treatment with JAK inhibitors. The case report describes a 73-year-old male with refractory BP that was successfully managed with the combination therapy of tofacitinib and low-dose glucocorticoids for 28 weeks. Immunohistochemistry and RNA sequencing were performed to analyze the underlying mechanism of tofacitinib therapy. A systematic literature search was conducted to identify other cases of treatment with JAK inhibitors. Throughout the 28-week treatment period, the patient experienced clinical, autoantibody and histologic resolution. Immunohistochemical analysis showed tofacitinib significantly decreased the pSTAT3 and pSTAT6 levels in the skin lesions of this patient. RNA sequencing and immunohistochemical testing of lesion samples from other BP patients identified activation of the JAK-STAT signaling pathway. Literature review revealed 17 previously reported cases of BP treated with four kinds of JAK inhibitors successfully, including tofacitinib (10), baricitinib (1), upadacitinib (3) and abrocitinib (3). Our findings support the potential of tofacitinib as a safe and effective treatment option for BP. Larger studies are underway to better understand this efficacy and safety.
2024-11-13 | GSE278926 | GEO
Project description:Background: Valproic acid (VPA) has been approved for the treatment of seizure disorders. It is also commonly used in psychiatric disorders, such as schizophrenia spectrum disorders. With increasing administration, reports of intoxications are more frequently reported. The most common findings of VPA intoxication are central nervous system depression, respiratory depression, hypotension, metabolic acidosis, and elevated lactate, among others. Methods: We describe a case report of VPA intoxication with hemodiafiltration (HDF) as extracorporeal treatment (ECTR) for removal of VPA. This treatment modality has only rarely been reported in the current literature. In addition, we performed an updated systematic literature review (SLR) of additional cases on the topic ranging from December 1st, 2014 to April 20th, 2018. We searched MEDLINE and Web of Science for relevant references. Results: In the presented case, VPA intoxication occurred in a 46-year-old female patient after oral ingestion of 56 g of VPA. In addition to vasopressors and endotracheal intubation, we administered L-Carnitine (L-Car) and performed hemodiafiltration treatment. After intravenous therapy with L-Car and simultaneous HDF sessions, we observed full recovery without neurological sequelae. The SLR identified 8 additional articles reporting favorable outcomes with extracorporeal treatments in most cases. Conclusion: HDF and other extracorporeal procedures are safe and effective therapeutic options in patients with VPA intoxication. The choice of ECTR modality mainly depends on local experience and the setting. In the present case, ingestion of 56 g was successfully treated with HDF. These findings are in line with several other case reports describing positive outcomes. Extracorporeal treatment, including HDF, should be considered early in the management of VPA intoxication. Supporting evidence is emerging, but it is of limited quality.
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Project description:IntroductionA compartment syndrome (CS) occurs when increased pressure within an anatomic compartment leads to inadequate perfusion. Although rare, gluteal CS can be encountered when an unconscious person has a prolonged period of immobilization.Presentation of caseA 20-year-old male with history of polysubstance abuse leading to passing out, presented with right buttock and lower extremity pain, increased creatinine phosphokinase (CPK), and acute renal failure. Physical examination and MRI confirmation supported gluteal CS. Patient was taken to the OR for gluteal fasciotomy. Afterwards, the pain improved, the CPK and creatinine trended to normal. He was discharged home on day 7.DiscussionCS can occur in any part of the body with fascial compartments. Increased compartmental pressure causes compression of vessels and nerves in the area that can lead to ischemia and necrosis. CS can occur after trauma, excessive fluid resuscitation, or surgery. It is also reported due to the prolonged periods of immobilization and increasing pressure on dependent areas. Often, intra-compartmental pressure is measured to confirm the diagnosis. The mainstay of treatment is fasciotomy.ConclusionDue to the rarity of gluteal compartment syndrome, the diagnosis is often delayed. If the affected area is ischemic for a significant amount of time, it can lead to sciatic nerve palsy, paresthesias, paralysis and muscle necrosis. Patients may experience irreversible damage after the syndrome and as such providers should be cognizant of this clinical entity to make an early diagnosis of gluteal compartment syndrome.
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Project description:BackgroundDrug reaction with eosinophilia and systemic symptoms (DRESS) is a severe hypersensitivity drug reaction involving the skin and multiple internal organ systems. The symptoms typically present with fever and skin rash, and rapidly progress to multiple organ failures. Vancomycin is a rare drug to cause DRESS syndrome with 23 cases reported to date.Case presentationWe described a case of a 39 year-old man who was treated with vancomycin for osteomyelitis of the foot. The patient subsequently developed acute respiratory distress syndrome (ARDS) followed by rash and acute interstitial nephritis. These symptoms were improved by withdrawal of vancomycin and a pulsed corticosteroid regimen. According to the European Registry of Severe Cutaneous Adverse Reaction Criteria (RegiSCAR) (Kardaun et al, British Journal of Dermatology, 169:1071-1080, 2013), the probability of vancomycin induced DRESS syndrome was scored as "Definite". A literature search of vancomycin induced DRESS syndrome was also performed and the overall pulmonary involvement was estimated as 5%. To our knowledge, this was the first case reported with pulmonary involvement as the initial symptom.ConclusionThis is the first case to report pulmonary manifestation as the initial symptom in vancomycin induced DRESS syndrome. Prompt recognition of this entity can expedite proper treatment and hasten recovery.
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Project description:Background: One of the most serious complications of cranial radiotherapy is the development of radiation-induced glioma, which is estimated to occur in 1 to 4% of patients who have received cranial irradiation and has a worse prognosis than sporadic glioblastoma. To date, owing to its rarity, no standard of care has been established for radiation-induced glioma. Although comprehensive genetic analysis has recently uncovered the molecular characteristics of radiation-induced glioma, the full picture remains unclear, and the molecular features associated with treatment response and prognosis are poorly understood. Case presentation: A 45-year-old man presented with generalized seizures caused by multiple brain tumors involving the right frontal lobe, thalamus, and brainstem. The patient had a history of whole-brain radiotherapy for the recurrence of Burkitt's lymphoma at the age of 12. He underwent craniotomy, and the histological diagnosis was a high-grade glioma with isocitrate dehydrogenase-wildtype, which was presumed to be a radiation-induced glioma that developed 33 years after whole-brain irradiation. The Heidelberg DNA-methylation brain classifier most closely matched diffuse pediatric-type high-grade glioma, receptor tyrosine kinase-1 subtype, which is a typical methylation class of radiation-induced glioma. Methylation-specific polymerase chain reaction showed that the O6-methylguanine-DNA methyltransferase gene promoter was unmethylated. Next-generation sequencing identified CDKN2A/B deletion as well as co-amplification of several receptor tyrosine kinase-encoding genes including PDGFRA, KIT, and KDR, which are all located on chromosome 4q12. Amplification of this region is present broadly across cancers and is associated with a poor prognosis in sporadic glioblastoma. Nevertheless, the patient received conventional chemoradiotherapy with temozolomide. Subsequent multimodal imaging with magnetic resonance imaging and 11C-methionine positron emission tomography revealed complete remission of all lesions. Two years later, the patient is currently alive with a favorable performance status. Conclusions: Despite radiation-induced glioma with molecular features suggestive of an aggressive phenotype, our patient unexpectedly responded well to conventional chemoradiotherapy, resulting in complete remission that is exceptional in sporadic glioblastoma. Our case indicates that some of the radiation-induced gliomas may have distinct molecular characteristics involved in the therapeutic response that differ from those of sporadic glioblastomas.
2025-05-14 | GSE276059 | GEO
Project description:IntroductionSalbutamol is a moderately selective beta-2-adrenergic agonist. Various side effects can occur because of beta-1 and beta-2 receptor activation. Due to the large volume of distribution, it is not considered dialyzable.Case presentationA patient with salbutamol intoxication, which developed as a result of a medical error in a patient with sepsis, Down syndrome, and liver cirrhosis, is presented. Initial treatment was partially successful and antibiotic adjustments were made. After his respiratory failure worsened, the patient needed non-invasive ventilation, and previously undiagnosed chronic obstructive pulmonary disease was suspected. He was prescribed intravenous methylprednisolone but accidently received 5 mg of salbutamol (albuterol), which led to immediate severe arrhythmic tachycardia with hemodynamic collapse. After unsuccessful cardioversion and treatment with landiolol infusion, salvage hemodialysis was commenced to decrease suspectedly highly elevated serum salbutamol levels. After 30 min, sinus rhythm with normocardia was observed. After the hemodialysis termination, no rebound tachycardia was noted, but due to severe septic shock, the hypotension was ongoing and vasoactive medications were adjusted. However, the measured levels of plasma salbutamol and data from literature do not support the view that hemodialysis was the cause of the described improvement: the total amount of the drug cleared was very small (2.8% of total dose).ConclusionOur results confirm a large volume of salbutamol distribution; the measured levels are within observed therapeutic levels; and the measured half-life time during hemodialysis (3.1 h) is comparable to observed half-life times in therapeutic settings. The observed favorable clinical benefit associated with dialysis may be fortuitous, highlighting potential bias toward positive clinical outcomes and unproven ("salvage") therapies.
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2017-03-24 | GSE96980 | GEO
Project description:BackgroundChylothorax as part of the clinical spectrum of tuberculosis (TB) is a rare entity, especially among children. However, it is crucial for clinicians to be able to identify, correlate, and diagnose chylothorax as it poses significant morbidity to patients.Case descriptionWe report on a paediatric case of pleural TB complicated with complex chylothorax, and systematically reviewing the literature for cases of tuberculous chylothorax among children particularly looking at the (I) demographic, (II) clinical presentations, (III) radiological findings, and (IV) investigations among this cohort of patients. The systematic review complied to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines. We used three separate databases, namely PubMed, Ovid, and Scopus to search for articles with terms "chylothorax", "tuberculosis", and "children". We included article reporting on: (I) children aged below 18; (II) infected by Mycobacterium TB, and (III) written in English only. We reviewed the publications from the inception up to August 2022. After an extensive search, 7 articles were reviewed. We included 10 patients from all the reports. Most common symptoms reported were intermittent fever (n=6) and chronic cough (n=5). Other symptoms include shortness of breath and chest discomfort due to fluid accumulation and/or effusion. Typical TB symptoms like night sweat, loss of weight, and loss of appetite were present in some of the reported patients. Chest radiography was the most used diagnostic imaging modality, in which all of the reported cases noted presence of pleural effusion, with some cases proceeded with computer tomography (CT) of thorax to aid in the diagnosis. Most of the patients had lymphadenopathies and all patients underwent pleural tapping for analysis and symptomatic relief. From the pleural fluid milky appearance and biochemistry correlation, the diagnosis of chylothorax was established in all 10 patients. Every patient was tested positive for TB infection.ConclusionsMost of the cases showed similar clinical presentation, radiological findings, and laboratory investigations particularly the classic analysis findings of pleural fluid. Even though TB is not a common cause of chylothorax, clinicians should have raised suspicion of it especially after gathering full history and correlating it with the other findings.
| S-EPMC10416126 | biostudies-literature
Project description:BackgroundTopiramate is an anticonvulsant from sulfamate-substituted monosaccharides that is increasingly used to treat migraines. Serious topiramate intoxications have been described. Unfortunately, indications for and the effect of interventions, including hemodialysis, in severe intoxications seem expert-based and lack empirical evidence. We aim to review the literature on topiramate intoxication cases and to describe the first topiramate intoxication with toxicokinetic data following treatment with hemodialysis.MethodsA literature review was conducted using the PubMed database. Included articles were reviewed for symptoms; management, including acute hemodialysis; toxicokinetic data; and outcomes.ResultsWe found 61 hits in the PubMed database and checked 392 references in the snowball search; 22 were included for data extraction, reporting 29 cases. The majority of the patients were female (n = 23/29, 79%), ranging in age from 2 to 44 years (median 21). The ingested topiramate amount ranged from 175 to 40,000 mg (usual maintenance dose of 50 mg BID and a general maximum of 500 mg BID). Topiramate concentrations were reported in eight cases, ranging from 3.7 to 356.6 mg/L (for reference, the therapeutic range is 2-30 mg/L). Serious topiramate intoxications can result in seizures, coma, hemodynamic instability and severe metabolic acidosis. In no single case was hemodialysis used.ConclusionSerious symptoms of topiramate intoxications exist, and hemodialysis is used infrequently. If symptoms are refractory to symptomatic treatment, hemodialysis can reduce topiramate concentrations and symptomatology.
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