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1.
Brain Spine ; 4: 102819, 2024.
Article in English | MEDLINE | ID: mdl-38706801

ABSTRACT

Introduction: Paediatric cerebellar glioblastoma is an exceptionally rare clinical entity, with very few cases described in the literature. In the majority of reported cases, prognosis is extremely poor, despite surgical and oncological management. The paucity of data results in lack of consensus as to the optimal management of these patients, with the objective of prolonging survival. Research question: Do patient or tumour characteristics suggest more favourable rates of progression-free survival in paediatric cerebellar glioblastoma? Material and methods: Tumour histopathology plus retrospective molecular analysis of archived samples, as well treatment strategy and patient characteristics of a six-year-old child with cerebellar glioblastoma and prolonged progression-free survival were assessed. Characteristics in the published literature that inferred prolonged survival were identified and compared. Results: Paediatric cerebellar glioblastoma is extremely rare, with only a handful of cases reported over several decades, during which time diagnostic and therapeutic techniques have evolved markedly. Consequently, the scarcity of data with sufficient granularity means that limited conclusions can be drawn. Specific clinical and histopathological factors (i.e. female sex, young age, EGFR negativity and surgical resection plus adjuvant chemoradiotherapy) may indicate a more favourable progression-free survival. Discussion and conclusion: Rates of progression-free survival in this rare condition are generally poor, however, several patient and tumour characteristics may infer more favourable prognosis. As increasingly refined means of diagnosis and characterisation are developed, particularly as a result of advances in molecular analyses, more adjuvant treatment options are likely to come on stream in future.

2.
Br J Neurosurg ; : 1-7, 2021 Feb 04.
Article in English | MEDLINE | ID: mdl-33538207

ABSTRACT

BACKGROUND: Pituitary dysfunction (PD) in granulomatosis with polyangiitis (GPA) is a rare manifestation of the disease with an incidence of 2% and tends to occur as the disease progresses. On very rare occasions, PD can be the initial presentation of GPA. From our literature review, only 6 such cases were documented. CASE PRESENTATION: We present a rare case of GPA in a 57-year-old female who primarily presented with PD and visual disturbance without any other systemic features of GPA. Her initial ANCA screen was negative and her MRI scan showed an enlarged pituitary gland with cystic changes. A pituitary biopsy confirmed the diagnosis and she was started on immunosuppressants. After completing her immunotherapy, her diabetic insipidus subsided and her vision markedly improved. LITERATURE REVIEW: A literature search on Pubmed, Cochrane, Open Grey, and SciELO with keywords 'granulomatosis', 'polyangiitis', and 'pituitary' was performed. A total of 72 articles were reviewed. 6 articles were identified where PD was the presenting complaint of GPA without other systemic involvement. DISCUSSION: Secondary hypophysitis in GPA indicates pituitary inflammation resulting from systemic disease or pituitary near-lying lesions. PD in GPA tends to occur with concomitant active disease at other sites. Headache seemed to be a common presenting feature with diabetic insipidus being the most common type of PD. Tissue biopsy is essential to confirm diagnosis. Pituitary biopsy is rarely required as other organ involvement is available for safer tissue sampling. However, in our case, pituitary biopsy had a role in confirming diagnosis and avoiding treatment delay. CONCLUSION: PD as a primary manifestation in GPA without systemic involvement is very rare and poses a diagnostic challenge. Pituitary biopsy can aid in diagnosis, allowing prompt treatment and prevention of long-term complications of untreated GPA such as permanent pituitary dysfunction. Nevertheless, the risk and benefit of pituitary surgery in each case should be weighted.

3.
Future Healthc J ; 6(Suppl 2): 65, 2019 Jun.
Article in English | MEDLINE | ID: mdl-31572957
4.
Sci Rep ; 9(1): 5885, 2019 04 10.
Article in English | MEDLINE | ID: mdl-30971773

ABSTRACT

The Dex-CSDH trial is a randomised, double-blind, placebo-controlled trial of dexamethasone for patients with a symptomatic chronic subdural haematoma. The trial commenced with an internal pilot, whose primary objective was to assess the feasibility of multi-centre recruitment. Primary outcome data collection and safety were also assessed, whilst maintaining blinding. We aimed to recruit 100 patients from United Kingdom Neurosurgical Units within 12 months. Trial participants were randomised to a 2-week course of dexamethasone or placebo in addition to receiving standard care (which could include surgery). The primary outcome measure of the trial is the modified Rankin Scale at 6 months. This pilot recruited ahead of target; 100 patients were recruited within nine months of commencement. 47% of screened patients consented to recruitment. The primary outcome measure was collected in 98% of patients. No safety concerns were raised by the independent data monitoring and ethics committee and only five patients were withdrawn from drug treatment. Pilot trial data can inform on the design and resource provision for substantive trials. This internal pilot was successful in determining recruitment feasibility. Excellent follow-up rates were achieved and exploratory outcome measures were added to increase the scientific value of the trial.


Subject(s)
Dexamethasone/therapeutic use , Hematoma, Subdural, Chronic/drug therapy , Dexamethasone/adverse effects , Double-Blind Method , Drug Administration Schedule , Hematoma, Subdural, Chronic/pathology , Humans , Pilot Projects , Placebo Effect , Randomized Controlled Trials as Topic , Treatment Outcome
6.
Trials ; 19(1): 670, 2018 12 04.
Article in English | MEDLINE | ID: mdl-30514400

ABSTRACT

BACKGROUND: Chronic subdural haematoma (CSDH) is a common neurosurgical condition, typically treated with surgical drainage of the haematoma. However, surgery is associated with mortality and morbidity, including up to 20% recurrence of the CSDH. Steroids, such as dexamethasone, have been identified as a potential therapy for reducing recurrence risk in surgically treated CSDHs. They have also been used as a conservative treatment option, thereby avoiding surgery altogether. The hypothesis of the Dex-CSDH trial is that a two-week course of dexamethasone in symptomatic patients with CSDH will lead to better functional outcome at six months. This is anticipated to occur through reduced number of hospital admissions and surgical interventions. METHODS: Dex-CSDH is a UK multi-centre, double-blind randomised controlled trial of dexamethasone versus placebo for symptomatic adult patients diagnosed with CSDH. A sample size of 750 patients has been determined, including an initial internal pilot phase of 100 patients to confirm recruitment feasibility. Patients must be recruited within 72 h of admission to a neurosurgical unit and exclusions include patients already on steroids or with steroid contraindications, patients who have a cerebrospinal fluid shunt and those with a history of psychosis. The decision regarding surgical intervention will be made by the clinical team and patients can be included in the trial regardless of whether operative treatment is planned or has been performed. The primary outcome measure is the modified Rankin Scale (mRS) at six months. Secondary outcomes include the number of CSDH-related surgical interventions during follow-up, length of hospital stay, mRS at three months, EQ-5D at three and six months, adverse events, mortality and a health-economic analysis. DISCUSSION: This multi-centre trial will provide high-quality evidence as to the effectiveness of dexamethasone in the treatment of CSDH. This has implications for patient morbidity and mortality as well as a potential economic impact on the overall health service burden from this condition. TRIAL REGISTRATION: ISRCTN, ISRCTN80782810 . Registered on 7 November 2014. EudraCT, 2014-004948-35 . Registered on 20 March 2015. Dex-CSDH trial protocol version 3, 27 Apr 2017. This protocol was developed in accordance with the SPIRIT checklist. Available as a separate document on request.


Subject(s)
Dexamethasone/administration & dosage , Glucocorticoids/administration & dosage , Hematoma, Subdural, Chronic/drug therapy , Cost-Benefit Analysis , Dexamethasone/adverse effects , Dexamethasone/economics , Double-Blind Method , Drug Administration Schedule , Drug Costs , Glucocorticoids/adverse effects , Glucocorticoids/economics , Hematoma, Subdural, Chronic/diagnosis , Hematoma, Subdural, Chronic/economics , Hematoma, Subdural, Chronic/mortality , Humans , Multicenter Studies as Topic , Randomized Controlled Trials as Topic , Time Factors , Treatment Outcome , United Kingdom
7.
Arthroscopy ; 30(9): 1166-72, 2014 Sep.
Article in English | MEDLINE | ID: mdl-24813322

ABSTRACT

PURPOSE: A systematic review was performed to assess the outcomes and complications of ultrasound-guided barbotage (repeated injection and aspiration) for calcific tendonitis of the shoulder. METHODS: A literature search of the Medline, Embase, and Cochrane databases using all relevant keywords found 1,454 original articles. After removal of duplicates and application of inclusion criteria, 13 original articles were selected for review. Articles that used fluoroscopic guidance rather than ultrasound guidance were excluded from the review. All studies analyzed except 1 were case series, with no comparative studies being available. RESULTS: Thirteen articles with a total of 908 patients were analyzed. In all articles reviewed, the authors reported a good clinical outcome, with many achieving marked improvement in clinical scores or overall satisfaction with the treatment. CONCLUSIONS: Ultrasound-guided barbotage is a safe technique, with a high success rate and low complication rate. There is no evidence assessing its effectiveness compared with other major treatment modalities; a randomized trial comparing ultrasound-guided barbotage, extracorporeal shock wave therapy, and arthroscopic calcific deposit excision would be of great value. However, while awaiting such a trial, on the basis of the results of this systematic review, we can recommend ultrasound-guided barbotage. LEVEL OF EVIDENCE: Level IV, systematic review of Level IV studies.


Subject(s)
Calcinosis/therapy , Tendinopathy/therapy , Ultrasonography, Interventional/methods , Female , Humans , Male , Middle Aged , Shoulder , Shoulder Pain/drug therapy , Suction/methods , Therapeutic Irrigation/methods
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