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Kavelin Rumalla, Kyle A. Smith, and Paul M. Arnold

Immunoglobulin G4–related disease (IgG4-RD) is a recently defined condition characterized by inflammatory tumefactive lesions in various organ systems. IgG4-RD is a clinical and radiological diagnosis of exclusion and requires the presence of specific histopathological criteria for diagnosis.

A 50-year-old man presented to an outside hospital with a 3-month history of progressively worsening back pain and symptoms of pleurisy, nasal crusting, and hematochezia. Radiological workup revealed an epidural-paraspinal mass with displacement of the spinal cord, destruction of the T5–6 vertebrae, and extension into the right lung. Biopsy sampling and subsequent histopathological analysis revealed dense lymphoplasmacytic infiltrate with an increased number of IgG4-positive plasma cells and a storiform pattern of fibrosis. With strong histopathological evidence of IgG4-RD, the patient was started on a regimen of prednisone. Further testing ruled out malignant neoplasm, infectious etiologies, and other autoimmune diseases. Two weeks later, the patient presented with acute-onset paraplegia due to spinal cord compression. The patient underwent decompression laminectomy of T5–6, posterior instrumented fusion of T2–8, and debulking of the epidural-paraspinal mass. After the continued administration of glucocorticosteroids, the patient improved remarkably to near-normal strength in the lower extremities and sensory function 6 months after surgery.

To the authors' knowledge, this is the first case of IgG4-related epidural inflammatory pseudotumor and spinal cord compression in the United States. This case highlights the importance of early administration of glucocorticosteroids, which were essential to preventing further progression and preventing relapse. IgG4-RD evaluation is important after other diseases in the differential diagnosis are ruled out.

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Kavelin Rumalla, Chester K. Yarbrough, Andrew J. Pugely, Linda Koester, and Ian G. Dorward

OBJECTIVE

The objective of this study was to determine if the recent changes in technology, surgical techniques, and surgical literature have influenced practice trends in spinal fusion surgery for pediatric neuromuscular scoliosis (NMS). In this study the authors analyzed recent trends in the surgical management of NMS and investigated the effect of various patient and surgical factors on in-hospital complications, outcomes, and costs, using the Nationwide Inpatient Sample (NIS) database.

METHODS

The NIS was queried from 2002 to 2011 using International Classification of Diseases, Ninth Edition, Clinical Modification codes to identify pediatric cases (age < 18 years) of spinal fusion for NMS. Several patient, surgical, and short-term outcome factors were included in the analyses. Trend analyses of these factors were conducted. Both univariate and multivariable analyses were used to determine the effect of the various patient and surgical factors on short-term outcomes.

RESULTS

Between 2002 and 2011, a total of 2154 NMS fusion cases were identified, and the volume of spinal fusion procedures increased 93% from 148 in 2002 to 286 in 2011 (p < 0.0001). The mean patient age was 12.8 ± 3.10 years, and 45.6% of the study population was female. The overall complication rate was 40.1% and the respiratory complication rate was 28.2%. From 2002 to 2011, upward trends (p < 0.0001) were demonstrated in Medicaid insurance status (36.5% to 52.8%), presence of ≥ 1 comorbidity (40.2% to 52.1%), and blood transfusions (25.2% to 57.3%). Utilization of posterior-only fusions (PSFs) increased from 66.2% to 90.2% (p < 0.0001) while combined anterior release/fusions and PSF (AR/PSF) decreased from 33.8% to 9.8% (< 0.0001). Intraoperative neurophysiological monitoring (IONM) underwent increasing utilization from 2009 to 2011 (15.5% to 20.3%, p < 0.0001). The use/harvest of autograft underwent a significant upward trend between 2002 and 2011 (31.3% to 59.8%, p < 0.0001). In univariate analysis, IONM use was associated with decreased complications (40.7% to 33.1%, p = 0.049) and length of stay (LOS; 9.21 to 6.70 days, p <0.0001). Inflation-adjusted mean hospital costs increased nearly 75% from 2002 to 2011 ($36,805 to $65,244, p < 0.0001). In the multivariable analysis, nonwhite race, highest quartile of median household income, greater preexisting comorbidity, long-segment fusions, and use of blood transfusions were found to increase the likelihood of complication occurrence (all p < 0.05). In further multivariable analysis, independent predictors of prolonged LOS included older age, increased preexisting comorbidity, the AR/PSF approach, and long-segment fusions (all p < 0.05). Lastly, the likelihood of increased hospital costs (at or above the 90th percentile for LOS, 14 days) was increased by older age, female sex, Medicaid insurance status, highest quartile of median household income, AR/PSF approach, long-segment fusion, and blood transfusion (all p < 0.05). In multivariable analysis, the use of autograft was associated with a lower likelihood of complication occurrence and prolonged LOS (both p < 0.05).

CONCLUSIONS

Increasing use of IONM and posterior-only approaches may combat the high complication rates in NMS. The trends of increasing comorbidities, blood transfusions, and total costs in spinal fusion surgery for pediatric NMS may indicate an increasingly aggressive approach to these cases.

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Malte Ottenhausen, Kavelin Rumalla, Iyan Younus, Shlomo Minkowitz, Apostolos John Tsiouris, and Theodore H. Schwartz

OBJECTIVE

Resection of supratentorial meningiomas is generally considered a low-risk procedure, but tumors involving the rolandic cortex present a unique challenge. The rate of motor function deterioration associated with resecting such tumors is not well described in the literature. Thus, the authors sought to report the rates and predictors of postoperative motor deficit following the resection of rolandic meningiomas to assist with patient counseling and surgical decision-making.

METHODS

An institution’s pathology database was screened for meningiomas removed between 2000 and 2017, and patients with neuroradiological evidence of rolandic involvement were identified. Parameters screened as potential predictors included patient age, sex, preoperative motor severity, tumor location, tumor origin (falx vs convexity), histological grade, FLAIR signal (T2-weighted MRI), venous involvement (T1-weighted MRI with contrast), intratumoral hemorrhage, embolization, and degree of resection (Simpson grade). Variables of interest included preoperative weakness and postoperative motor decline (novel or worsened permanent deficit). The SPSS univariate and bivariate analysis functions were used, and statistical significance was determined with alpha < 0.05.

RESULTS

In 89 patients who had undergone resection of convexity (80.9%) or parasagittal (19.1%) rolandic meningiomas, a postoperative motor decline occurred in 24.7%. Of 53 patients (59.6%) with preoperative motor deficits, 60.3% improved, 13.2% were unchanged, and 26.4% worsened following surgery. Among the 36 patients without preoperative deficits, 22.2% developed new weakness. Predictors of preoperative motor deficit included tumor size (41.6 vs 33.2 cm3, p = 0.040) and presence of FLAIR signal (69.8% vs 50.0%, p = 0.046). Predictors of postoperative motor decline were preoperative motor deficit (47.2% vs 22.2%, p = 0.017), minor (compared with severe) preoperative weakness (25.6% vs 21.4%, p < 0.001), and preoperative embolization (54.5% vs 20.5%, p = 0.014). Factors that trended toward significance included parafalcine tumor origin (41.2% vs 20.8% convexity, p = 0.08), significant venous involvement (44.4% vs 23.5% none, p = 0.09), and Simpson grade II+ (34.2% vs 17.6% grade I, p = 0.07).

CONCLUSIONS

Resection of rolandic area meningiomas carries a high rate of postoperative morbidity and deserves special preoperative planning. Large tumor size, peritumoral edema, preoperative embolization, parafalcine origin, and venous involvement may further increase the risk. Alternative surgical strategies, such as aggressive internal debulking, may prevent motor decline in a subset of high-risk patients.

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Joshua S. Catapano, Kavelin Rumalla, Visish M. Srinivasan, Peter M. Lawrence, Kristen Larson Keil, and Michael T. Lawton

OBJECTIVE

Brainstem cavernous malformations (BSCMs) are complex, difficult to access, and highly variable in size, shape, and position. The authors have proposed a novel taxonomy for pontine cavernous malformations (CMs) based upon clinical presentation (syndromes) and anatomical location (findings on MRI).

METHODS

The proposed taxonomy was applied to a 30-year (1990–2019), 2-surgeon experience. Of 601 patients who underwent microsurgical resection of BSCMs, 551 with appropriate data were classified on the basis of BSCM location: midbrain (151 [27%]), pons (323 [59%]), and medulla (77 [14%]). Pontine lesions were then subtyped on the basis of their predominant surface presentation identified on preoperative MRI. Neurological outcomes were assessed according to the modified Rankin Scale, with a score ≤ 2 defined as favorable.

RESULTS

The 323 pontine BSCMs were classified into 6 distinct subtypes: basilar (6 [1.9%]), peritrigeminal (53 [16.4%]), middle peduncular (MP) (100 [31.0%]), inferior peduncular (47 [14.6%]), rhomboid (80 [24.8%]), and supraolivary (37 [11.5%]). Part 1 of this 2-part series describes the taxonomic basis for the first 3 of these 6 subtypes of pontine CM. Basilar lesions are located in the anteromedial pons and associated with contralateral hemiparesis. Peritrigeminal lesions are located in the anterolateral pons and are associated with hemiparesis and sensory changes. Patients with MP lesions presented with mild anterior inferior cerebellar artery syndrome with contralateral hemisensory loss, ipsilateral ataxia, and ipsilateral facial numbness without cranial neuropathies. A single surgical approach and strategy were preferred for each subtype: for basilar lesions, the pterional craniotomy and anterior transpetrous approach was preferred; for peritrigeminal lesions, extended retrosigmoid craniotomy and transcerebellopontine angle approach; and for MP lesions, extended retrosigmoid craniotomy and trans–middle cerebellar peduncle approach. Favorable outcomes were observed in 123 of 143 (86%) patients with follow-up data. There were no significant differences in outcomes between the 3 subtypes or any other subtypes.

CONCLUSIONS

The neurological symptoms and key localizing signs associated with a hemorrhagic pontine subtype can help to define that subtype clinically. The proposed taxonomy for pontine CMs meaningfully guides surgical strategy and may improve patient outcomes.

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Joshua S. Catapano, Kavelin Rumalla, Visish M. Srinivasan, Peter M. Lawrence, Kristen Larson Keil, and Michael T. Lawton

OBJECTIVE

Part 2 of this 2-part series on pontine cavernomas presents the taxonomy for subtypes 4–6: inferior peduncular (IP) (subtype 4), rhomboid (5), and supraolivary (6). (Subtypes 1–3 are presented in Part 1.) The authors have proposed a novel taxonomy for pontine cavernous malformations based on clinical presentation (syndromes) and anatomical location (MRI findings).

METHODS

The details of taxonomy development are described fully in Part 1 of this series. In brief, pontine lesions (323 of 601 [53.7%] total lesions) were subtyped on the basis of predominant surface presentation identified on preoperative MRI. Neurological outcomes were assessed according to the modified Rankin Scale, with score ≤ 2 defined as favorable.

RESULTS

The 323 pontine brainstem cavernous malformations were classified into 6 distinct subtypes: basilar (6 [1.9%]), peritrigeminal (53 [16.4%]), middle peduncular (100 [31.0%]), IP (47 [14.6%]), rhomboid (80 [24.8%]), and supraolivary (37 [11.5%]). Subtypes 4–6 are the subject of the current report. IP lesions are located in the inferolateral pons and are associated with acute vestibular syndrome. Rhomboid lesions present to the fourth ventricle floor and are associated with disconjugate eye movements. Larger lesions may cause ipsilateral facial weakness. Supraolivary lesions present to the surface at the ventral pontine underbelly. Ipsilateral abducens palsy is a strong localizing sign for this subtype. A single surgical approach and strategy were preferred for subtypes 4–6: for IP cavernomas, the suboccipital craniotomy and telovelar approach predominated; for rhomboid lesions, the suboccipital craniotomy and transventricular approach were preferred; and for supraolivary malformations, the far lateral craniotomy and transpontomedullary sulcus approach were preferred. Favorable outcomes were observed in 132 of 150 (88%) patients with follow-up. There were no significant differences in outcomes between subtypes.

CONCLUSIONS

The neurological symptoms and signs associated with a hemorrhagic pontine subtype can help define that subtype clinically with key localizing signs. The proposed taxonomy for pontine cavernous malformation subtypes 4–6 meaningfully guides surgical strategy and may improve patient outcomes.

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Kavelin Rumalla, Visish M. Srinivasan, Monica Gaddis, Peter Kan, Michael T. Lawton, and Jan-Karl Burkhardt

OBJECTIVE

Extracranial-intracranial (EC-IC) bypass surgery remains an important treatment option for patients with moyamoya disease (MMD), intracranial arteriosclerotic disease (ICAD) with symptomatic stenosis despite the best medical management, and complex aneurysms. The therapeutic benefit of cerebral bypass surgery depends on optimal patient selection and the minimization of periprocedural complications. The nationwide burden of readmissions and associated complications following EC-IC bypass surgery has not been previously described. Therefore, the authors sought to analyze a nationwide database to describe the national rates, causes, risk factors, complications, and morbidity associated with readmission following EC-IC bypass surgery for MMD, ICAD, and aneurysms.

METHODS

The Nationwide Readmissions Database (NRD) was queried for the years 2010–2014 to identify patients who had undergone EC-IC bypass for MMD, medically failed symptomatic ICAD, or unruptured aneurysms. Predictor variables included demographics, preexisting comorbidities, indication for surgery, and hospital bypass case volume. A high-volume center (HVC) was defined as one that performed 10 or more cases/year. Outcome variables included perioperative stroke, discharge disposition, length of stay, total hospital costs, and readmission (30 days, 90 days). Multivariable analysis was used to identify predictors of readmission and to study the effect of treatment at HVCs on quality outcomes.

RESULTS

In total, 2500 patients with a mean age of 41 years were treated with EC-IC bypass surgery for MMD (63.1%), ICAD (24.5%), or unruptured aneurysms (12.4%). The 30- and 90-day readmission rates were 7.5% and 14.0%, respectively. Causes of readmission included new stroke (2.5%), wound complications (2.5%), graft failure (1.5%), and other infection (1.3%). In the multivariable analysis, risk factors for readmission included Medicaid/self-pay (OR 1.6, 95% CI 1.1–2.4, vs private insurance), comorbidity score (OR 1.2, 95% CI 1.1–1.4, per additional comorbidity), and treatment at a non-HVC (OR 1.9, 95% CI 1.1–3.0). Treatment at an HVC (17% of patients) was associated with significantly lower rates of nonroutine discharge dispositions (13.4% vs 26.7%, p = 0.004), ischemic stroke within 90 days (0.8% vs 2.9%, p = 0.03), 30-day readmission (3.9% vs 8.2%, p = 0.03), and 90-day readmission (8.6% vs 15.2%, p = 0.01). These findings were confirmed in a multivariable analysis. The authors estimate that centralization to HVCs may result in 333 fewer nonroutine discharges (50% reduction), 12,000 fewer hospital days (44% reduction), 165 fewer readmissions (43%), and a cost savings of $15.3 million (11% reduction).

CONCLUSIONS

Readmission rates for patients after EC-IC bypass are comparable with those after other common cranial procedures and are primarily driven by preexisting comorbidities, socioeconomic status, and treatment at low-volume centers. Periprocedural complications, including stroke, graft failure, and wound complications, occurred at the expected rates, consistent with those in prior clinical series. The centralization of care may significantly reduce perioperative complications, readmissions, and hospital resource utilization.

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Joshua S. Catapano, Kavelin Rumalla, Visish M. Srinivasan, Peter M. Lawrence, Kristen Larson Keil, and Michael T. Lawton

OBJECTIVE

Medullary cavernous malformations are the least common of the brainstem cavernous malformations (BSCMs), accounting for only 14% of lesions in the authors’ surgical experience. In this article, a novel taxonomy for these lesions is proposed based on clinical presentation and anatomical location.

METHODS

The taxonomy system was applied to a large 2-surgeon experience over a 30-year period (1990–2019). Of 601 patients who underwent microsurgical resection of BSCMs, 551 were identified who had the clinical and radiological information needed for inclusion. These 551 patients were classified by lesion location: midbrain (151 [27%]), pons (323 [59%]), and medulla (77 [14%]). Medullary lesions were subtyped on the basis of their predominant surface presentation. Neurological outcomes were assessed according to the modified Rankin Scale (mRS), with an mRS score ≤ 2 defined as favorable.

RESULTS

Five distinct subtypes were defined for the 77 medullary BSCMs: pyramidal (3 [3.9%]), olivary (35 [46%]), cuneate (24 [31%]), gracile (5 [6.5%]), and trigonal (10 [13%]). Pyramidal lesions are located in the anterior medulla and were associated with hemiparesis and hypoglossal nerve palsy. Olivary lesions are found in the anterolateral medulla and were associated with ataxia. Cuneate lesions are located in the posterolateral medulla and were associated with ipsilateral upper-extremity sensory deficits. Gracile lesions are located outside the fourth ventricle in the posteroinferior medulla and were associated with ipsilateral lower-extremity sensory deficits. Trigonal lesions in the ventricular floor were associated with nausea, vomiting, and diplopia. A single surgical approach was preferred (> 90% of cases) for each medullary subtype: the far lateral approach for pyramidal and olivary lesions, the suboccipital-telovelar approach for cuneate lesions, the suboccipital-transcisterna magna approach for gracile lesions, and the suboccipital-transventricular approach for trigonal lesions. Of these 77 patients for whom follow-up data were available (n = 73), 63 (86%) had favorable outcomes and 67 (92%) had unchanged or improved functional status.

CONCLUSIONS

This study confirms that the constellation of neurological signs and symptoms associated with a hemorrhagic medullary BSCM subtype is useful for defining the BSCM clinically according to a neurologically recognizable syndrome at the bedside. The proposed taxonomical classifications may be used to guide the selection of surgical approaches, which may enhance the consistency of clinical communications and help improve patient outcomes.

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Kavelin Rumalla, Visish M. Srinivasan, Monica Gaddis, Peter Kan, Michael T. Lawton, and Jan-Karl Burkhardt

OBJECTIVE

Extracranial-intracranial (EC-IC) bypass surgery remains an important treatment option for patients with moyamoya disease (MMD), intracranial arteriosclerotic disease (ICAD) with symptomatic stenosis despite the best medical management, and complex aneurysms. The therapeutic benefit of cerebral bypass surgery depends on optimal patient selection and the minimization of periprocedural complications. The nationwide burden of readmissions and associated complications following EC-IC bypass surgery has not been previously described. Therefore, the authors sought to analyze a nationwide database to describe the national rates, causes, risk factors, complications, and morbidity associated with readmission following EC-IC bypass surgery for MMD, ICAD, and aneurysms.

METHODS

The Nationwide Readmissions Database (NRD) was queried for the years 2010–2014 to identify patients who had undergone EC-IC bypass for MMD, medically failed symptomatic ICAD, or unruptured aneurysms. Predictor variables included demographics, preexisting comorbidities, indication for surgery, and hospital bypass case volume. A high-volume center (HVC) was defined as one that performed 10 or more cases/year. Outcome variables included perioperative stroke, discharge disposition, length of stay, total hospital costs, and readmission (30 days, 90 days). Multivariable analysis was used to identify predictors of readmission and to study the effect of treatment at HVCs on quality outcomes.

RESULTS

In total, 2500 patients with a mean age of 41 years were treated with EC-IC bypass surgery for MMD (63.1%), ICAD (24.5%), or unruptured aneurysms (12.4%). The 30- and 90-day readmission rates were 7.5% and 14.0%, respectively. Causes of readmission included new stroke (2.5%), wound complications (2.5%), graft failure (1.5%), and other infection (1.3%). In the multivariable analysis, risk factors for readmission included Medicaid/self-pay (OR 1.6, 95% CI 1.1–2.4, vs private insurance), comorbidity score (OR 1.2, 95% CI 1.1–1.4, per additional comorbidity), and treatment at a non-HVC (OR 1.9, 95% CI 1.1–3.0). Treatment at an HVC (17% of patients) was associated with significantly lower rates of nonroutine discharge dispositions (13.4% vs 26.7%, p = 0.004), ischemic stroke within 90 days (0.8% vs 2.9%, p = 0.03), 30-day readmission (3.9% vs 8.2%, p = 0.03), and 90-day readmission (8.6% vs 15.2%, p = 0.01). These findings were confirmed in a multivariable analysis. The authors estimate that centralization to HVCs may result in 333 fewer nonroutine discharges (50% reduction), 12,000 fewer hospital days (44% reduction), 165 fewer readmissions (43%), and a cost savings of $15.3 million (11% reduction).

CONCLUSIONS

Readmission rates for patients after EC-IC bypass are comparable with those after other common cranial procedures and are primarily driven by preexisting comorbidities, socioeconomic status, and treatment at low-volume centers. Periprocedural complications, including stroke, graft failure, and wound complications, occurred at the expected rates, consistent with those in prior clinical series. The centralization of care may significantly reduce perioperative complications, readmissions, and hospital resource utilization.

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Kavelin Rumalla, Kyle A. Smith, Vijay Letchuman, Mrudula Gandham, Rachana Kombathula, and Paul M. Arnold

OBJECTIVE

Posttraumatic seizures (PTSs) are the most common complication following a traumatic brain injury (TBI) and may lead to posttraumatic epilepsy. PTS is well described in the adult literature but has not been studied extensively in children. Here, the authors utilized the largest nationwide registry of pediatric hospitalizations to report the national incidence, risk factors, and outcomes associated with PTS in pediatric TBI.

METHODS

The authors queried the Kids’ Inpatient Database (KID) using ICD-9-CM codes to identify all patients (age < 21 years) who had a primary diagnosis of TBI (850.xx–854.xx) and a secondary diagnosis of PTS (780.33, 780.39). Parameters of interest included patient demographics, preexisting comorbidities, hospital characteristics, nature of injury (open/closed), injury type (concussion, laceration/contusion, subarachnoid hemorrhage, subdural hematoma, or epidural hematoma), loss of consciousness (LOC), surgical management (Clinical Classification Software code 1 or 2), discharge disposition, in-hospital complications, and in-hospital mortality. The authors utilized the IBM SPSS statistical package (version 24) for univariate comparisons, as well as the identification of independent risk factors for PTS in multivariable analysis (alpha set at < 0.05).

RESULTS

The rate of PTS was 6.9% among 124,444 unique patients hospitalized for TBI. The utilization rate of continuous electroencephalography (cEEG) was 0.3% and increased between 2003 (0.1%) and 2012 (0.7%). The most common etiologies of TBI were motor vehicle accident (n = 50,615), accidental fall (n = 30,847), and blunt trauma (n = 13,831). However, the groups with the highest rate of PTS were shaken infant syndrome (41.4%), accidental falls (8.1%), and cycling accidents (7.4%). In multivariable analysis, risk factors for PTS included age 0–5 years (compared with 6–10, 11–15, and 16–20 years), African American race (OR 1.4), ≥ 3 preexisting comorbidities (OR 4.0), shaken infant syndrome (OR 4.4), subdural hematoma (OR 1.6), closed-type injury (OR 2.3), brief LOC (OR 1.4), moderate LOC (OR 1.5), and prolonged LOC with baseline return (OR 1.8). Surgically managed patients were more likely to experience PTS (OR 1.5) unless they were treated within 24 hours of admission (OR 0.8). PTS was associated with an increased likelihood of in-hospital complications (OR 1.7) and adverse (nonroutine) discharge disposition (OR 1.2), but not in-hospital mortality (OR 0.5). The overall utilization rate of cEEG was 1.3% in PTS patients compared with 0.2% in patients without PTS. Continuous EEG monitoring was associated with higher rates of diagnosed PTS (35.4% vs 6.8%; OR 4.9, p < 0.001).

CONCLUSIONS

PTS is common in children with TBI and is associated with adverse outcomes. Independent risk factors for PTS include younger age (< 5 years), African American race, increased preexisting comorbidity, prolonged LOC, and injury pattern involving cortical exposure to blood products. However, patients who undergo urgent surgical evacuation are less likely to develop PTS.

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Kavelin Rumalla, Meic H. Schmidt, and Christian A. Bowers