The Management of Spontaneous Otogenic CSF Leaks: A Presentation of Cases and Review of Literature.
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ABSTRACT:
SUBMITTER: Wilson MN
PROVIDER: S-EPMC3969441 | biostudies-other | 2014 Apr
REPOSITORIES: biostudies-other
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Project description:ObjectiveTo review the literature and our institutional experience regarding the risk of meningitis in patients with spontaneous lateral skull base cerebrospinal fluid (sCSF) leaks awaiting surgical repair, and the roles of antibiotic prophylaxis and pneumococcal vaccination, if known.MethodsA retrospective chart review and systematic review of the literature was undertaken to identify the incidence of meningitis in patients with sCSF leaks awaiting surgical repair. Adults managed surgically for sCSF leaks at an academic tertiary care center over a 10-year period were included. Data was collected on receipt of prophylactic antibiotics and/or pneumococcal vaccines during the timeframe between diagnosis and surgical repair.ResultsInstitutional review identified 87 patients who underwent surgical repair of spontaneous leaks, with a 0% incidence of meningitis over a median duration of 2 months while awaiting surgery (mean 5.5 months, range 0.5-118 months). Eighty-eight percent of patients did not receive prophylactic antibiotics. No studies in the published literature demonstrated the impact of prophylactic antibiotics or pneumococcal vaccine on meningitis risk.ConclusionsThere appears to be a low risk of meningitis among patients with lateral skull base sCSF leaks awaiting surgery for short durations (≤2 months), even in the absence of prophylactic antibiotics. There is a substantial gap in the published literature assessing the risk of meningitis and roles of antibiotics and vaccination in this patient population, indicating the need for large-scale study to conclusively elucidate the nature of this risk.
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Project description:Introduction ?Cerebrospinal fluid (CSF) rhinorrhea is the leakage of CSF through nasal cavity, due to abnormal communication between the arachnoid membrane and nasal mucosa. Middle-age (fourth to fifth decade) group, female gender, and obesity (body mass index?>?40) are the most commonly reported risk-factors for this rare entity. In this study, we present our single center experience of spontaneous CSF rhinorrhea discussing important clinicoradiological aspects in preoperative evaluation and nuances in the endoscopic repair technique. Material and Methods ?A retrospective study conducted for 43 spontaneous CSF rhinorrhea patients admitted between Jan 2011 to Jan 2018 at our tertiary care center. All patients underwent endoscopic repair of the defect depending upon their site of leak. Results ?Mean age in our study was 36.7?±?12.3 years (range: 9-62 years). Average BMI in males was found lower (28.7) as compared with females (32). Most common site of CSF leak was cribriform plate ( n ?=?32, 74.4%) and Planum was found to be the least common site ( n ?=?1, 2.3%) of CSF leak. Intraoperatively, 23 (53.5%) patients showed high-flow leak. Intrathecal injection of fluorescein dye was used to identify the site of CSF leakage in 15 cases (34.8%). The overall success rate of primary endoscopic repair in our study was 95.3%. Conclusion ?Spontaneous CSF rhinorrhea occurs secondary to elevated intracranial pressure, with a predilection for obese females in fourth to fifth decade. Individualized tailored surgical approach depending upon the site, size, and flow-variety of the defect forms the cornerstone of management.
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Project description:The pathophysiology of giant cerebral aneurysms renders them difficult to treat. Advances in technology have attempted to address any shortcomings associated with open surgery or endovascular therapies. Since the introduction of the flow diversion technique, the endovascular approach with flow diversion has become the first-line modality chosen to treat giant aneurysms. A subset of these giant aneurysms may persistent despite any treatment modality. Perhaps the best option for these recurrent and/or persistent giant aneurysms is to employ a multimodal approach-both surgical and endovascular-rather than any single technique to provide a curative result with favourable patient outcomes. This paper provides a review of the histopathology and treatment options for giant cerebral aneurysms. Additionally, an illustrative case is presented to highlight the unique challenges of a curative solution for giant cerebral aneurysms that persist despite initial treatment.
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Project description:Gray platelet syndrome (GPS) is a rare hereditary hemorrhagic disorder characterized by macrothrombocytopenia and the absence of alpha-granules in platelets. Clinically, mild-to-moderate bleeding is the main manifestation, often accompanied by thrombocytopenia, splenomegaly, and myelofibrosis. Here, we present a case of a 15-year-old male patient with a history of hepatosplenomegaly, and thrombocytopenia for 8 years, who presented with sudden generalized abdominal pain. Despite initial suspicion of gastroenteritis, diagnostic imaging revealed an extensive hemoperitoneum. Subsequent genetic testing confirmed the diagnosis of GPS, which had not been previously identified. This case highlights the importance of considering inherited platelet disorders should be considered in adolescents with long-standing thrombocytopenia, and emphasizes the need for thorough evaluation in patients with suggestive symptoms.
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Project description:In patients with occult cerebrospinal fluid (CSF) leaks or CSF leak syndrome, orthostatic headaches are a common presenting symptom. Although computed tomography (CT) myelography has historically been the gold standard for diagnosis with radioisotope cisternography as a diagnostic alternative, magnetic resonance imaging (MRI) myelography using intrathecal gadolinium has reported sensitivity of 80%-87%. Two patients with spontaneous orthostatic headaches lasting for several days were diagnosed with CSF leaks at multiple thoracic segments using MRI myelogram with intrathecal gadolinium (Gadavist, Bayer, Whippany, NJ). This allowed for subsequent targeted treatment with CT fluoroscopy guidance, resulting in therapeutic responses within 1-2 treatment with targeted blood patching. Although intrathecal gadolinium is an off-label use, the superior contrast resolution and lack of radiation exposure makes MRI myelography an excellent imaging modality for diagnosing CSF leak, targeting treatment, and monitoring outcomes compared to CT myelography and radioisotope cisternography.
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Project description:Chromoblastomycosis and leprosy are chronic diseases with high prevalence in tropical and subtropical regions. Brazil is one of the countries with the highest incidence and prevalence for both diseases, however, reports of co-infections are scarce. The aim of this study was to describe three cases of chromoblastomycosis-leprosy co-infection in patients from Mato Grosso state, Brazil. A review of chromoblastomycosis-leprosy co-infection was performed of English, Portuguese and Spanish publications in LILACS, SciELO, PubMed and Web of Science databases using the descriptors (chromoblastomycosis OR cromoblastomicose OR cromoblastomicosis) AND (leprosy OR hanseníase OR lepra), without time period delimitation. Nineteen cases were included, 16 cases were published in 11 articles, plus the three cases reported in the current study. Most reported coninfection cases came from Brazil. Majority of the patients were male with a mean age of 52.2 years. Farmer was the main occupational activity reported. In 12 patients, the clinical signs and symptoms of leprosy started first. No contacts with patients affected by leprosy, armadillos or history of injuries at the anatomical site of chromoblastomycosis lesions were reported. Five leprosy patients who received steroid treatment for leprosy reactions or neuropathies, were diagnosed with chromoblastomycosis during immunosuppressive therapy. Four cases (21.1%) were reported among the elderly patients. Co-infections in patients with chromoblastomycosis or leprosy are uncommon, but the possibility should always be considered, especially if the patient is undergoing immunosuppressive treatment or is elder.
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Project description:Upper gastrointestinal perforations, leaks and fistulae are relatively common occurrences with a growing number of these complications occuring as a result of therapeutic advancement and adoption of newer and bolder endoscopic therapies. Historically, these were predominantly managed surgically; however, owing to high morbidity and mortality associated with surgical repair, endoscopic options are preferable. Over the past decade, vast expansion in the endoscopic armamentarium for the management of perforations, leaks and fistulae has led to endoscopic management now being the first-line treatment. Here, we will review the endoscopic modalities including through-the-scope clips, over-the-scope clips, stents, vacuum therapy, endoscopic sutures and sealants. In addition, we will discuss nonendoscopic approach to management including early recognition of perforations, ways to reduce septic complications and format algorithms to guide therapy for different scenarios. However, it is important to stress that there is a lack of high-quality randomised studies to clearly guide management of such complications, resulting in a wide variation of approaches in management by specialists. Each case requires some degree of individualisation due to the potential array of problems encountered and patient-specific co-morbidities. In the future, more robust studies are clearly required to better guide specialist management.
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Project description:BACKGROUND:N-Acetylglutamate synthase (NAGS) deficiency is an extremely rare autosomal recessive metabolic disorder affecting the urea cycle, leading to episodes of hyperammonemia which can cause significant morbidity and mortality. Since its recognition in 1981, NAGS deficiency has been treated with carbamylglutamate with or without other measures (nutritional, ammonia scavengers, dialytic, etc.). We conducted a systematic literature review of NAGS deficiency to summarize current knowledge around presentation and management. METHODS:Case reports and case series were identified using the Medline database, as well as references from other articles and a general internet search. Clinical data related to presentation and management were abstracted by two reviewers. RESULTS:In total, 98 cases of NAGS deficiency from 79 families, in 48 articles or abstracts were identified. Of these, 1 was diagnosed prenatally, 57 were neonatal cases, 34 were post-neonatal, and 6 did not specify age at presentation or were asymptomatic at diagnosis. Twenty-one cases had relevant family history. We summarize triggers of hyperammonemic episodes, diagnosis, clinical signs and symptoms, and management strategies. DNA testing is the preferred method of diagnosis, although therapeutic trials to assess response of ammonia levels to carbamylglutamate may also be helpful. Management usually consists of treatment with carbamylglutamate, although the reported maintenance dose varied across case reports. Protein restriction was sometimes used in conjunction with carbamylglutamate. Supplementation with citrulline, arginine, and sodium benzoate also were reported. CONCLUSIONS:Presentation of NAGS deficiency varies by age and symptoms. In addition, both diagnosis and management have evolved over time and vary across clinics. Prompt recognition and appropriate treatment of NAGS deficiency with carbamylglutamate may improve outcomes of affected individuals. Further research is needed to assess the roles of protein restriction and supplements in the treatment of NAGS deficiency, especially during times of illness or lack of access to carbamylglutamate.
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Project description:BackgroundChyle leakage is an uncommon but potentially life-threatening complication following esophageal resections. The optimal management strategy is not clear, with a limited evidence base.MethodsSearches were conducted up to 31 December 2020 on MEDLINE, Embase, and Web of Science for randomized trials or retrospective studies that evaluated the management of chyle leakage following esophageal resection. Two authors independently screened studies, extracted data, and assessed for bias. The protocol was prospectively registered on PROSPERO (CRD: 42021224895) and reported in accordance with preferred reporting items for systematic reviews and meta-analyses guidelines.ResultsA total of 530 citations were reviewed. Twenty-five studies, totaling 1016 patients met the inclusion criteria, including two low-quality clinical trials and 23 retrospective case series. Heterogeneity of study design and outcomes prevented meta-analysis. The overall incidence of chyle leak/fistula was 3.2%. Eighteen studies describe management of chyle leaks conservatively, 17 by surgical ligation of the thoracic duct, 5 by pleurodesis, and 6 described percutaneous lymphangiography with thoracic duct embolization or disruption.ConclusionsThe evidence base for optimal management of chyle leakage postesophagectomy is lacking, which may be related to its low incidence. There is a paucity of high-quality prospective studies directly comparing treatment modalities, but there is some low-certainty evidence that percutaneous approaches have reduced morbidity but lower efficacy compared with surgery. Further high-quality, prospective studies that compare interventions at different levels of severity are needed to determine the optimal approach to treatment.
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Project description:Patients with a combination of holoprosencephaly and polydactyly, but with apparently normal chromosomes, may be clinically diagnosed with holoprosencephaly-polydactyly syndrome (HPS), also termed pseudotrisomy 13. However, the criteria for HPS have been controversial since the advent of the diagnostic term, and a clear understanding of the condition lacks definitive delineation. We review the historical and current perspectives on the condition and analyze findings in 40 patients with apparent HPS, including cases from the literature and two previously unreported patients. Overall, our analysis suggests previously unrecognized trends in patients diagnosed with HPS. Specifically, there appears to be a higher prevalence of visceral anomalies, most significantly cardiac and genitourinary, but also with increased gastrointestinal, pulmonary, adrenal, skeletal, and renal abnormalities, in patients with HPS. Although these visceral anomalies may not be essential for the identification of HPS, clinicians should be aware of the presence of such characteristics in these patients to optimize management and help establish etiologies.
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