ANCA-associated kidney disease preceded by orbital pseudotumor.

INTRODUCTION: Antineutrophil cytoplasmic antibody (ANCA)-associated vasculitis (AAV) and IgG4-related disease (IgG4-RD) are distinct immune disorders with overlapping clinical and laboratory features. While ANCA positivity excludes IgG4-RD in the 2019 ACR/EULAR classification, this criterion is not uniformly applied, and AAV can form inflammatory masses in various organs and show increase in IgG4 + plasma cells, similar to IgG4-RD. CASE DIAGNOSIS/TREATMENT: A 5-year-old female with history of orbital mass diagnosed as IgG4-RD presents with acute kidney injury. She has a myeloperoxidase ANCA, and kidney biopsy shows pauci-immune crescentic glomerulonephritis and acute tubulointerstitial nephritis with increased IgG4 + plasma cells and tubular basement membrane (TBM) deposits. CONCLUSION: In isolation, TBM deposits and increased IgG4 + plasma cells are suggestive of IgG4-RD. In the context of a positive ANCA and pauci-immune crescentic glomerulonephritis, however, increased IgG4 + plasma cells due to AAV are favored. In cases with features of IgG4-RD, ANCA positivity suggests an alternate diagnosis of AAV to be more likely.

Combination Resective or Ablative Epilepsy Surgery with Neurostimulation for Complex Epilepsy Networks: A Case Series.

INTRODUCTION: Complex epilepsy networks with multifocal onset zones that overlap with eloquent cortex may benefit from combined surgical approaches. However, limited data exist on outcomes associated with performing these therapies in tandem. In this case series, we report on 6 patients who underwent combination surgery with either resection or laser interstitial thermal therapy (LITT) and neuromodulation with responsive neurostimulation (RNS) or deep brain stimulation (DBS). METHODS: We performed a retrospective review of adult patients with medically refractory epilepsy who underwent staged combination epilepsy surgeries during the same admission at our institution. Six cases treated between 2019 and 2023 were identified. All patients underwent a presurgical work-up including invasive intracranial monitoring and underwent a combined approach with either surgical resection, LITT, RNS, or DBS. We extracted data on demographic, clinical, and surgical characteristics. The primary outcome was change in seizure frequency from baseline. RESULTS: The mean age was 42.7 years old (4 female). All patients had at least one epileptogenic zone in the temporal lobe, two in extratemporal neocortex, two in periventricular nodular heterotopia. For the staged combination approach, 3 patients underwent LITT followed by RNS, two underwent resection and RNS, and one received LITT and DBS. The mean reduction in seizure frequency per month at last follow-up was 90%. Postoperatively, 1 patient experienced superior visual field deficits related to LITT, and another had postoperative deep vein thrombosis. CONCLUSION: All patients experienced at least an 83% reduction in seizures. This case series demonstrates the potential benefits of a combined surgical approach in patients with multifocal seizures and at least one lesion that can be safely resected or ablated. Future prospective studies are warranted.

Taste dysfunction after endoscopic endonasal resection of olfactory groove meningioma: Case series and review of the literature.

PURPOSE: The incidence of ageusia and dysgeusia after endoscopic endonasal (EEA) resection of olfactory groove meningioma (OGM) is not well established despite recognized impairment in olfactory function. METHODS: We retrospectively administered a validated taste and smell survey to patients undergoing EEA for resection of OGM at two institutions. Demographics and clinical characteristics were collected and survey responses were analyzed. RESULTS: Twelve patients completed the survey. The median time from surgery was 24 months. The average total complaint score was 5.5 out of 16 [0-13]. All patients reported a change in sense of smell while only 42 % reported a change in sense of taste. Taste changes did not consistently associate with laterality or size of the neoplasm. Significant heterogeneity existed when rating severity of symptoms. CONCLUSIONS: To our knowledge this is the first case series examining taste changes after EEA resection of OGM. Despite universal olfactory dysfunction, only a minority of patients reported a change in their sense of taste. Our findings may improve patient counseling and expectations after surgery.

Beta Oscillations in the Sensory Thalamus During Severe Facial Neuropathic Pain Using Novel Sensing Deep Brain Stimulation.

OBJECTIVE: Advancements in deep brain stimulation (DBS) devices provide a unique opportunity to record local field potentials longitudinally to improve the efficacy of treatment for intractable facial pain. We aimed to identify potential electrophysiological biomarkers of pain in the ventral posteromedial nucleus (VPM) of the thalamus and periaqueductal gray (PAG) using a long-term sensing DBS system. MATERIALS AND METHODS: We analyzed power spectra of ambulatory pain-related events from one patient implanted with a long-term sensing generator, representing different pain intensities (pain >7, pain >9) and pain qualities (no pain, burning, stabbing, and shocking pain). Power spectra were parametrized to separate oscillatory and aperiodic features and compared across the different pain states. RESULTS: Overall, 96 events were marked during a 16-month follow-up. Parameterization of spectra revealed a total of 62 oscillatory peaks with most in the VPM (77.4%). The pain-free condition did not show any oscillations. In contrast, ß peaks were observed in the VPM during all episodes (100%) associated with pain >9, 56% of episodes with pain >7, and 50% of burning pain events (center frequencies: 28.4 Hz, 17.8 Hz, and 20.7 Hz, respectively). Episodes of pain >9 indicated the highest relative ß band power in the VPM and decreased aperiodic exponents (denoting the slope of the power spectra) in both the VPM and PAG. CONCLUSIONS: For this patient, an increase in ß band activity in the sensory thalamus was associated with severe facial pain, opening the possibility for closed-loop DBS in facial pain.

Probabilistic modeling reveals coordinated social interaction states and their multisensory bases.

Social behavior across animal species ranges from simple pairwise interactions to thousands of individuals coordinating goal-directed movements. Regardless of the scale, these interactions are governed by the interplay between multimodal sensory information and the internal state of each animal. Here, we investigate how animals use multiple sensory modalities to guide social behavior in the highly social zebrafish (Danio rerio) and uncover the complex features of pairwise interactions early in development. To identify distinct behaviors and understand how they vary over time, we developed a new hidden Markov model with constrained linear-model emissions to automatically classify states of coordinated interaction, using the movements of one animal to predict those of another. We discovered that social behaviors alternate between two interaction states within a single experimental session, distinguished by unique movements and timescales. Long-range interactions, akin to shoaling, rely on vision, while mechanosensation underlies rapid synchronized movements and parallel swimming, precursors of schooling. Altogether, we observe spontaneous interactions in pairs of fish, develop novel hidden Markov modeling to reveal two fundamental interaction modes, and identify the sensory systems involved in each. Our modeling approach to pairwise social interactions has broad applicability to a wide variety of naturalistic behaviors and species and solves the challenge of detecting transient couplings between quasi-periodic time series.

Myxopapillary Ependymoma Metastasis Mimicking Pulmonary Embolism: An Illustrative Case.

Myxopapillary ependymomas (MPEs) are rare spinal cord tumors with low rates of metastasis outside of the neuraxis. Gross total resection of MPEs can significantly improve progression-free survival; however, adjunctive treatment remains unstandardized. A 29-year-old female with a history of spina bifida occulta surgical correction and lower back pain presented with dyspnea and tachycardia. A large pulmonary artery mass was discovered consistent with pulmonary thromboembolism. It was subsequently determined to be an intravascular metastasis secondary to sacral MPE. Standardization of MPE treatment and clinical suspicion of spinal neoplasm in the setting of chronic back pain with undetermined origin are of value.

Probabilistic modeling reveals coordinated social interaction states and their multisensory bases.

Social behavior across animal species ranges from simple pairwise interactions to thousands of individuals coordinating goal-directed movements. Regardless of the scale, these interactions are governed by the interplay between multimodal sensory information and the internal state of each animal. Here, we investigate how animals use multiple sensory modalities to guide social behavior in the highly social zebrafish (Danio rerio) and uncover the complex features of pairwise interactions early in development. To identify distinct behaviors and understand how they vary over time, we developed a new hidden Markov model with constrained linear-model emissions to automatically classify states of coordinated interaction, using the movements of one animal to predict those of another. We discovered that social behaviors alternate between two interaction states within a single experimental session, distinguished by unique movements and timescales. Long-range interactions, akin to shoaling, rely on vision, while mechanosensation underlies rapid synchronized movements and parallel swimming, precursors of schooling. Altogether, we observe spontaneous interactions in pairs of fish, develop novel hidden Markov modeling to reveal two fundamental interaction modes, and identify the sensory systems involved in each. Our modeling approach to pairwise social interactions has broad applicability to a wide variety of naturalistic behaviors and species and solves the challenge of detecting transient couplings between quasi-periodic time series.

On-Call Workload Differences in Neurosurgery: Resident Call "Weather" and a Departure from Colored Cloud Labels.

BACKGROUND AND OBJECTIVES: Labeling residents as "black" or "white" clouds based on perceived or presumed workloads is a timeworn custom across medical training and practice. Previous studies examining whether such perceptions align with objective workload patterns have offered conflicting results. We assessed whether such peer-assigned labels were associated with between-resident differences in objective, on-call workload metrics in three classes of neurosurgery junior residents. In doing so, we introduce more inclusive terminology for perceived differences in workload metrics. METHODS: Residents were instructed to complete surveys to identify "sunny", "neutral", and "stormy" residents, reflecting least to greatest perceived workloads, of their respective classes. We retrospectively reviewed department and electronic medical records to record volume of on-call work over the first 4 months of each resident's 2nd postgraduate academic year. Inter-rater agreement of survey responses was measured using Fleiss' kappa. All statistical analyses were performed with a significance threshold of P < .05. RESULTS: Across all classes, there was strong inter-rater agreement in the identification of stormy and sunny residents (Kappa = 1.000, P = .003). While differences in on-call workload measures existed within each class, "weather" designations did not consistently reflect these differences. There were significant intraclass differences in per shift consult volume in two classes ( P = .035 and P = .009); however, consult volume corresponded to a resident's weather designations in only one class. Stormy residents generally saw more emergencies and, in 2 classes, performed more bedside procedures than their peers. CONCLUSION: Significant differences in objective on-call experience exist between junior neurosurgery residents. Self- and peer-assigned weather labels did not consistently align with a pattern of these differences, suggesting that other factors contribute to such labels.

Rapid Passive Gamma Mapping as an Adjunct to Electrical Stimulation Mapping for Functional Localization in Resection of Primary Brain Neoplasms.

OBJECTIVE: We examined the utility of passive high gamma mapping (HGM) as an adjunct to conventional awake brain mapping during glioma resection. We compared functional and survival outcomes before and after implementing intraoperative HGM. METHODS: This was a retrospective cohort study of 75 patients who underwent a first-time, awake craniotomy for glioma resection. Patients were stratified by whether their operation occurred before or after the implementation of a U.S. Food and Drug Administration-approved high-gamma mapping tool in July 2017. RESULTS: The preimplementation and postimplementation cohorts included 28 and 47 patients, respectively. Median intraoperative time (261 vs. 261 minutes, P = 0.250) and extent of resection (97.14% vs. 98.19%, P = 0.481) were comparable between cohorts. Median Karnofsky performance status at initial follow-up was similar between cohorts (P = 0.650). Multivariable Cox regression models demonstrated an adjusted hazard ratio for overall survival of 0.10 (95% confidence interval: 0.02-0.43, P = 0.002) for the postimplementation cohort relative to the preimplementation cohort. Progression-free survival adjusted for insular involvement showed an adjusted hazard ratio of 1.00 (95% confidence interval: 0.49-2.06, P = 0.999) following HGM implementation. Falling short of statistical significance, prevalence of intraoperative seizures and/or afterdischarges decreased after HGM implementation as well (12.7% vs. 25%, P = 0.150). CONCLUSIONS: Our results tentatively indicate that passive HGM is a safe and potentially useful adjunct to electrical stimulation mapping for awake cortical mapping, conferring at least comparable functional and survival outcomes with a nonsignificant lower rate of intraoperative epileptiform events. Considering the limitations of our study design and patient cohort, further investigation is needed to better identify optimal use cases for HGM.

Low-Cost Wound Healing Protocol Reduces Infection and Reoperation Rates After Cranioplasty: A Retrospective Cohort Study.

BACKGROUND AND OBJECTIVES: Cranioplasty infections are a common and expensive problem associated with significant morbidity. Our objective was to determine whether a wound healing protocol after cranioplasty reduced the rate of infections and to determine the value of this intervention. METHODS: This is a single-institution retrospective chart review of 2 cohorts of cranioplasty patients over 12 years. The wound healing protocol, consisting of vitamin and mineral supplementation, fluid supplementation, and oxygen support, was instituted for all patients aged older than 15 years undergoing cranioplasty. We retrospectively reviewed the charts of all patients over the study period and compared outcomes before and after protocol institution. Outcomes included surgical site infection, return to operating room within 30 days, and cranioplasty explant. Cost data were collected from the electronic medical record. We included 291 cranioplasties performed before the wound healing protocol and 68 postprotocol. RESULTS: Baseline demographics and comorbidities were comparable between preprotocol and postprotocol groups. Odds of takeback to operating room within 30 days were the same before and after the wound healing protocol (odds ratio [OR] 2.21 [95% CI 0.76-6.47], P = .145). Odds of clinical concern for surgical site infection were significantly higher in the preprotocol group (OR 5.21 [95% CI 1.22-22.17], P = .025). Risk of washout was higher in the preprotocol group (HR 2.86 [95% CI 1.08-7.58], P = .035). Probability of cranioplasty flap explant was also significantly higher in the preprotocol group (OR 4.70 [95% CI 1.10-20.05], P = .036). The number needed to treat to prevent 1 cranioplasty infection was 24. CONCLUSION: A low-cost wound healing protocol was associated with reduced rate of infections after cranioplasty with concomitant reduction in reoperations for washout, saving the health care system more than $50,000 per 24 patients. Prospective study is warranted.

Low body mass index patients have worse outcomes after mechanical thrombectomy.

BACKGROUND: There is evidence that frailty is an independent predictor of worse outcomes after stroke. Similarly, although obesity is associated with a higher risk for stroke, there are multiple reports describing improved mortality and functional outcomes in higher body mass index (BMI) patients in a phenomenon known as the obesity paradox. We investigated the effect of low BMI on outcomes after mechanical thrombectomy (MT). METHODS: We conducted a retrospective analysis of 231 stroke patients who underwent MT at an academic medical center between 2020-2022. The patients' BMI data were collected from admission records and coded based on the Centers for Disease Control and Prevention (CDC) obesity guidelines. Recursive partitioning analysis (RPA) in R software was employed to automatically detect a BMI threshold associated with a significant survival benefit. Frailty was quantified using the Modified Frailty Index 5 and 11. RESULTS: In our dataset, by CDC classification, 2.6% of patients were underweight, 27.3% were normal BMI, 30.7% were overweight, 19.9% were class I obese, 9.5% were class II obese, and 10% were class III obese. There were no significant differences between these groups. RPA identified a clinically significant BMI threshold of 23.62 kg/m2. Independent of frailty, patients with a BMI ≤23.62 kg/m2 had significantly worse overall survival (P<0.001) and 90-day modified Rankin Scale (P=0.027) than patients above the threshold. CONCLUSIONS: Underweight patients had worse survival and functional outcomes after MT. Further research should focus on the pathophysiology underlying poor prognosis in underweight MT patients, and whether optimizing nutritional status confers any neuroprotective benefit.