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Rosai-Dorfman disease (RDD) with craniocervical junction involvement is a rare clinical entity. We present herein a case of a pediatric patient with craniocervical junction RDD which was surgically treated. A 10-year-old female with a history of B-cell acute lymphoblastic leukemia (B-ALL) in remission and RDD presented with frontal migraine headaches. She previously had a right posterior chest wall lesion which was biopsy-proven RDD. She was found on imaging to have a dural-based right craniocervical junction lesion. Given her history of B-ALL, after a multidisciplinary discussion, the decision was made to proceed with resection with possible initiation of cobimetinib or clofarabine. The patient underwent a suboccipital craniotomy, C1 laminectomy, and resection of the dural-based lesion. Gross total resection was achieved, and histopathology confirmed the diagnosis of RDD. She was discharged home on postoperative day 4. No recurrence was seen on follow-up imaging at 3 months. We conducted a systematic literature review examining all cases of pediatric intracranial RDD and all cases of craniocervical junction RDD. This represents, to the best of our knowledge, only the second case of pediatric craniocervical junction RDD. Although RDD is often self-limiting, medical treatment is often considered for intracranial disease, but tissue confirmation is necessary. Surgical resection provides histopathologic diagnosis and can sometimes serve as definitive treatment for a particular lesion.
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OBJECTIVES: To describe the mechanisms and visual outcomes of recreational and sports-related open globe injuries (OGIs). METHODS: A retrospective case series of eyes experiencing OGI secondary to recreational and sports-related activities at Memorial Hermann Hospital - Texas Medical Center (MHH-TMC) from January 1st, 2010 through March 31st, 2015 was conducted. Exclusionary criteria included no documented ophthalmologic examination upon presentation and repairs performed by services other than ophthalmology. A two-tailed t-test and Fisher's exact test were utilized to assess for statistical significance (p < 0.05). RESULTS: A total of 20 eyes from 20 patients experiencing OGIs secondary to recreational and sports-related activities were included. Thirteen eyes (65 %) presented with OGIs from penetrating objects while seven eyes (35 %) had injuries from blunt injuries. Males comprised most of the total study group (17 of 20 patients), and zone 3 injuries were the most common location of OGI. While eyes with OGIs from blunt trauma underwent evisceration/enucleation procedures more frequently than OGIs from penetrating mechanisms (71% vs 23 %) (p = 0.10), the final visual outcomes were similarly poor between groups. Only three eyes in this series experienced an improvement from baseline VA; all three eyes had lacked initial findings consistent with severe injury. CONCLUSIONS: Recreational and sports related OGIs most commonly occur in zone 3 and in young males, regardless of injury type. OGIs due to both penetrating and blunt trauma mechanisms implicate poor functional outcomes, but the absence of certain presenting injury features may indicate possibility of eventual visual recovery.
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Ferimentos Oculares Penetrantes , Traumatismos Oculares , Esportes , Ferimentos não Penetrantes , Masculino , Humanos , Feminino , Estudos Retrospectivos , Acuidade Visual , Traumatismos Oculares/complicações , Ferimentos não Penetrantes/complicações , PrognósticoRESUMO
INTRODUCTION: Injury and subsequent thrombosis of the cerebral venous sinuses may be caused by closed head injuries secondary to a variety of different mechanisms. Skull fractures can lacerate or otherwise disrupt adjacent dural sinuses. The sequelae of such injuries may include thrombosis and either partial or total occlusion of the sinus, ultimately resulting in significant venous congestion. Sagittal sinus injury is associated with a more serious outcome due to the obligatory flow into the sinus, especially posterior to the coronal suture. In such cases, venous infarction may be a severe and life-threatening complication of head injury. CASE PRESENTATION: A 2-year-old female presented with a depressed skull fracture near the midline and a thrombus in the sagittal sinus. Anticoagulation, the standard treatment cerebral venous sinus thrombosis (CVST), was contraindicated due to intracranial hemorrhage, so immediate thrombectomy was performed with successful neurologic recovery at 9-month follow-up. To our knowledge, this case is the youngest patient documented to receive mechanical thrombectomy for superior sagittal sinus (SSS) thrombosis due to trauma. CONCLUSION: Closed head injuries in pediatric patients may be associated with CVST, with resulting venous drainage compromise and profound neurologic sequelae. Unlike adult patients with spontaneous CVST in which anticoagulation are the standard of care, pediatric patients experiencing traumatic CVST may have contraindications to anticoagulants. If the patient has a contraindication to anticoagulation such as intracranial bleeding, endovascular mechanical thrombectomy may be an effective intervention when performed by an experienced neurointerventionalist.
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Seio Sagital Superior , Trombectomia , Humanos , Feminino , Pré-Escolar , Trombectomia/métodos , Seio Sagital Superior/cirurgia , Seio Sagital Superior/lesões , Trombose do Seio Sagital/cirurgia , Trombose do Seio Sagital/etiologia , Trombose dos Seios Intracranianos/cirurgia , Trombose dos Seios Intracranianos/diagnóstico por imagem , Fratura do Crânio com Afundamento/cirurgia , Fratura do Crânio com Afundamento/diagnóstico por imagem , Traumatismos Craniocerebrais/complicações , Traumatismos Craniocerebrais/cirurgiaRESUMO
PURPOSE: Pituitary apoplexy can be a life threatening and vision compromising event. Antiplatelet and anticoagulation use has been reported as a contributing factor in pituitary apoplexy (PA). Utilizing one of the largest cohorts in the literature, this study aims to determine the risk of PA in patients on antiplatelet/anticoagulation (AP/AC) therapy. METHODS: A single center, retrospective study was conducted on 342 pituitary adenoma patients, of which 77 patients presented with PA (23%). Several potential risk factors for PA were assessed, including: patient demographics, tumor characteristics, pre-operative hormone replacement, neurologic deficits, coagulation studies, platelet count, and AP/AC therapy. RESULTS: Comparing patients with and without apoplexy, there was no significant difference in the proportion of patients taking aspirin (45 no apoplexy vs. 10 apoplexy; p = 0.5), clopidogrel (10 no apoplexy vs. 4 apoplexy; p = 0.5), and anticoagulation (7 no apoplexy vs. 3 apoplexy; p = 0.7). However, male sex (p-value < 0.001) was a predictor for apoplexy while pre-operative hormone treatment was a protective factor from apoplexy (p-value < 0.001). A non-clinical difference in INR was also noted as a predictor for apoplexy (no apoplexy: 1.01 ± 0.09, apoplexy: 1.07 ± 0.15; p < 0.001). CONCLUSIONS: Although pituitary tumors have a high risk for spontaneous hemorrhage, the use of aspirin is not a risk for hemorrhage. Our study did not find an increased risk of apoplexy with clopidogrel or anticoagulation, but further investigation is needed with a larger cohort. Confirming other reports, male sex is associated with an increased risk for PA.