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Amir Ahmadian, Jotham Manwaring, Devon Truong, Jeane McCarthy, Luis F. Rodriguez, Carolyn M. Carey and Gerald F. Tuite

Vascular access in the neonate can be challenging, especially in preterm infants. When other access is not available, superficial scalp veins can be safely used for vascular access. However, rare and potentially catastrophic complications can occur due to unique features of the neonatal skull and soft-tissue anatomy. The authors report a rare complication of vascular access in a preterm infant, which led to the direct infusion of parenteral nutrition into the intracranial space. The child had an excellent outcome after open drainage and irrigation of bilateral intracranial spaces and the spinal thecal sac. Relevant anatomy is illustrated, and an outcome-based literature review is presented on this rarely reported condition. Surgical and conservative management strategies are discussed, along with clinical and radiographic follow-up. Drainage and irrigation is advocated in patients with mass effect, viscous effusions, or declining neurological examination findings.

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Marta Ortega-Martínez, Jose M. Cabezudo, Ignacio Fernández-Portales, Manuel Pineda-Palomo, Jose Antonio Rodríguez-Sánchez and Luis Miguel Bernal-García

✓Hemangioblastomas are low-grade, highly vascular tumors commonly associated with von Hippel–Lindau (VHL) syndrome and most often appearing in the cerebellum. They very rarely occur in the spinal nerve roots, and an origin in the filum terminale is exceptional with no instances of multiple hemangioblastomas of the filum terminale reported in the literature. Because of their vascular nature, these lesions can enlarge and become symptomatic in the context of the changes that take place during pregnancy, as has been noted with cerebellar hemangioblastomas. In any case, the evolution of spinal hemangioblastomas during pregnancy is not well known given its rarity. The conjunction of both processes—that is, multiple hemangioblastomas arising in the filum terminale and pregnancy—is unique. The authors describe the case of a 41-year-old woman with multiple hemangioblastomas of the filum terminale and no other evidence of VHL syndrome, in whom pregnancy precipitated symptoms. The interruption of gestation led to a remission of the symptoms. The literature concerning filum terminale hemangioblastomas and pregnancy is also reviewed.

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Amir Ahmadian, Ali A. Baa j, Michael Garcia, Carolyn Carey, Luis Rodriguez, Bruce Storrs and Gerald F. Tuite

The authors present a case of extreme brain herniation encountered during decompressive craniectomy in a 21-month-old boy who suffered a trauma event that necessitated temporary scalp closure in which a sterile silicone sheet was placed. Although the clinical situation is usually expected to lead to brain death or severe disability, the patient's 3-year follow-up examination revealed a highly functional child with a good quality of life. The authors discuss the feasibility and advantages of temporary scalp expansion as a treatment option when extreme brain herniation is encountered during craniotomy.

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Andrew C. Vivas, Nir Shimony, Eric M. Jackson, Risheng Xu, George I. Jallo, Luis Rodriguez, Gerald F. Tuite and Carolyn M. Carey

OBJECTIVE

Hydrocephalus associated with subdural hygromas is a rare complication after decompression of Chiari malformation type I (CM-I). There is no consensus for management of this complication. The authors present a series of 5 pediatric patients who underwent CM-I decompression with placement of a dural graft complicated by posterior fossa hygromas and hydrocephalus that were successfully managed nonoperatively.

METHODS

A retrospective review over the last 5 years of patients who presented with hydrocephalus and subdural hygromas following foramen magnum decompression with placement of a dural graft for CM-I was conducted at 2 pediatric institutions. Their preoperative presentation, perioperative hospital course, and postoperative re-presentation are discussed with attention to their treatment regimen and ultimate outcome. In addition to reporting these cases, the authors discuss all similar cases found in their literature review.

RESULTS

Over the last 5 years, the authors have encountered 194 pediatric cases of CM-I decompression with duraplasty equally distributed at the 2 institutions. Of those cases, 5 pediatric patients with a delayed postoperative complication involving hydrocephalus and subdural hygromas were identified. The 5 patients were managed nonoperatively with acetazolamide and high-dose dexamethasone; dosages of both drugs were adjusted to the age and weight of each patient. All patients were symptom free at follow-up and exhibited resolution of their pathology on imaging. Thirteen similar pediatric cases and 17 adult cases were identified in the literature review. Most reported cases were treated with CSF diversion or reoperation. There were a total of 4 cases previously reported with successful nonoperative management. Of these cases, only 1 case was reported in the pediatric population.

CONCLUSIONS

De novo hydrocephalus, in association with subdural hygromas following CM-I decompression, is rare. This presentation suggests that these complications after posterior fossa decompression with duraplasty can be treated with nonoperative medical management, therefore obviating the need for CSF diversion or reoperation.

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Alberto Gil, Luis López-Ibor, Gerardo Lopez-Flores, Hugo Cuellar, Eduardo Murias and Gregorio Rodríguez-Boto

Endovascular treatment is the treatment of choice for indirect carotid cavernous fistulas (CCFs). Direct surgical obliteration of CCFs is recommended in highly symptomatic patients or in those with an aggressive pattern of venous drainage. However, this is a technically challenging approach associated with significant procedural morbidity. The authors present a case in which they decided to attempt a novel access to the cavernous sinus through the foramen ovale before recommending surgery for an otherwise untreatable dural CCF.

This 52-year-old man with an indirect CCF and neurological deficit had undergone several attempts to embolize the shunt by means of the standard approaches. Ultimately direct cavernous sinus access was obtained through the foramen ovale, resulting in complete obliteration of the shunt. The occlusion was radiographically stable at the 6-month follow-up evaluation, and the patient has remained asymptomatic. Percutaneous transovale puncture of a CCF is a feasible alternative to accessing the cavernous sinus when traditional transvenous catheterization or direct superior ophthalmic vein approach is not possible.

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Andrew S. Little, Luis Perez-Orribo, Nestor G. Rodriguez-Martinez, Phillip M. Reyes, Anna G. U. S. Newcomb, Daniel M. Prevedello and Neil R. Crawford

Object

Endoscopic endonasal approaches to the craniovertebral junction (CVJ) and clivus are increasingly performed for ventral skull-base pathology, but the biomechanical implications of these approaches have not been studied. The aim of this study was to investigate the spinal biomechanics of the CVJ after an inferior-third clivectomy and anterior intradural exposure of the foramen magnum as would be performed in an endonasal endoscopic surgical strategy.

Methods

Seven upper-cervical human cadaveric specimens (occiput [Oc]–C2) underwent nondestructive biomechanical flexibility testing during flexion-extension, axial rotation, and lateral bending at Oc–C1 and C1–2. Each specimen was tested intact, after an inferior-third clivectomy, and after ligamentous complex dissection simulating a wide intradural exposure using an anterior approach. Angular range of motion (ROM), lax zone, and stiff zone were determined and compared with the intact state.

Results

Modest, but statistically significant, hypermobility was observed after inferior-third clivectomy and intradural exposure during flexion-extension and axial rotation at Oc–C1. Angular ROM increased incrementally between 6% and 12% in flexion-extension and axial rotation. These increases were primarily the result of changes in the lax zone. No significant changes were noted at C1–2.

Conclusions

Inferior-third clivectomy and an intradural exposure to the ventral CVJ and foramen magnum resulted in hypermobility at Oc–C1 during flexion-extension and axial rotation. Although the results were statistically significant, the modest degree of hypermobility observed compared with other well-characterized CVJ injuries suggests that occipitocervical stabilization may be unnecessary for most patients.

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Stephen L. Reintjes, Ernest K. Amankwah, Luis F. Rodriguez, Carolyn C. Carey and Gerald F. Tuite

OBJECT

Fusion rates are high for children undergoing posterior cervical fusion (PCF) and occipito-cervical fusion (OCF). Autologous bone has been widely used as the graft material of choice, despite the risk of donor-site morbidity associated with harvesting the bone, possibly because very low fusion rates were reported with posterior allograft cervical fusions in children several decades ago. Higher overall fusion rates using allograft in adults, associated with improvements in internal fixation techniques and the availability of osteoinductive substances such as bone morphogenetic protein (BMP), have led to heightened enthusiasm for the use of bank bone during pediatric PCF. A systematic review was performed to study factors associated with successful bone fusion, including the type of bone graft used.

METHODS

The authors performed a comprehensive PubMed search of English-language articles pertaining to PCF and OCF in patients less than 18 years old. Of the 561 abstracts selected, 148 articles were reviewed, resulting in 60 articles that had sufficient detail to be included in the analysis. A meta-regression analysis was performed to determine if and how age, fusion technique, levels fused, fusion substrate, BMP use, postoperative bracing, and radiographic fusion criteria were related to the pooled prevalence estimates. A systematic review of the literature was performed according to the PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) statement.

RESULTS

A total of 604 patients met the specific inclusion and exclusion criteria. The overall fusion rate was 93%, with a mean age of 9.3 years and mean follow-up of 38.7 months. A total of 539 patients had fusion with autograft (94% fusion rate) and 65 patients with allograft (80% fusion rate). Multivariate meta-regression analysis showed that higher fusion rates were associated with OCF compared with fusions that excluded the occiput (p < 0.001), with the use of autograft instead of allograft (p < 0.001), and with the use of CT to define fusion instead of plain radiography alone. The type of internal fixation, the use of BMP, patient age, and the duration of follow-up were not found to be associated with fusion rates in the multivariate analysis.

CONCLUSIONS

Fusion rates for PCF are high, with higher rates of fusion seen when autograft is used as the bone substrate and when the occiput is included in the fusion construct. Further study of the use of allograft as a viable alternative to autograft bone fusion is warranted because limited data are available regarding the use of allograft in combination with more rigid internal fixation techniques and osteoinductive substances, both of which may enhance fusion rates with allograft.

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Samuel Browd, Lindsay J. Healy, Ginger Dobie, J. Thomas Johnson III, Greg M. Jones, Luis F. Rodriguez and Douglas L. Brockmeyer

Object

Congenital occipitocervical (OC) instability is uncommon in healthy children but can occur in many children with Down syndrome. A simple morphometric method of evaluating the OC joint in children with OC instability is presented, supported by a qualitative image analysis based on computed tomography (CT).

Methods

Thin-cut CT scans of the OC joint were obtained in eight patients with Down syndrome and one patient with congenital OC instability. These patients’ CT scans were compared with those of 15 healthy age-matched control individuals. Morphometric analysis was performed by measuring the depth and length of the superior articular surface (SAS) of C-1, and these values were normalized for a comparison between groups. Qualitative data were acquired using a surface-rendering technique for a visual comparison of the C-1 SAS.

Morphometric analysis demonstrated an absence of the concave C-1 SAS anatomy in patients with congenital OC instability compared with age-matched control individuals (0.083 compared with 0.202, p < 0.001). Three-dimensional (3D) image analysis of the C-1 SAS supported this finding.

Conclusions

Congenital differences in the shape of the OC joint are highly associated with atraumatic OC instability in children with Down syndrome. High-resolution CT imaging combined with 3D rendering techniques and surface mapping provides support for this assessment. It appears that abnormal OC joint shape is a contributing factor to congenital OC instability, especially in patients with Down syndrome.

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Nestor G. Rodriguez-Martinez, Luis Perez-Orribo, Samuel Kalb, Phillip M. Reyes, Anna G. U. S. Newcomb, Jeremy Hughes, Nicholas Theodore and Neil R. Crawford

OBJECT

The effects of obesity on lumbar biomechanics are not fully understood. The aims of this study were to analyze the biomechanical differences between cadaveric L4–5 lumbar spine segments from a large group of nonobese (body mass index [BMI] < 30 kg/m2) and obese (BMI ≥ 30 kg/m2) donors and to determine if there were any radiological differences between spines from nonobese and obese donors using MR imaging.

METHODS

A total of 168 intact L4–5 spinal segments (87 males and 81 females) were tested using pure-moment loading, simulating flexion-extension, lateral bending, and axial rotation. Axial compression tests were performed on 38 of the specimens. Sex, age, and BMI were analyzed with biomechanical parameters using 1-way ANOVA, Pearson correlation, and multiple regression analyses. MR images were obtained in 12 specimens (8 from obese and 4 from nonobese donors) using a 3-T MR scanner.

RESULTS

The segments from the obese male group allowed significantly greater range of motion (ROM) than those from the nonobese male group during axial rotation (p = 0.018), while there was no difference between segments from obese and nonobese females (p = 0.687). There were no differences in ROM between spines from obese and nonobese donors during flexion-extension or lateral bending for either sex. In the nonobese population, the ROM during axial rotation was significantly greater for females than for males (p = 0.009). There was no significant difference between sexes in the obese population (p = 0.892). Axial compressive stiffness was significantly greater for the obese than the nonobese population for both the female-only group and the entire study group (p < 0.01); however, the difference was nonsignificant in the male population (p = 0.304). Correlation analysis confirmed a significant negative correlation between BMI and resistance to deformation during axial compression in the female group (R = −0.65, p = 0.004), with no relationship in the male group (R = 0.03, p = 0.9). There was also a significant negative correlation between ROM during flexion-extension and BMI for the female group (R = −0.38, p = 0.001), with no relationship for the male group (R = 0.06, p = 0.58). Qualitative analysis using MR imaging indicated greater facet degeneration and a greater incidence of disc herniations in the obese group than in the control group.

CONCLUSIONS

Based on flexibility and compression tests, lumbar spinal segments from obese versus nonobese donors seem to behave differently, biomechanically, during axial rotation and compression. The differences are more pronounced in women. MR imaging suggests that these differences may be due to greater facet degeneration and an increased amount of disc herniation in the spines from obese individuals.

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Takao Hoshino, Luis A. Rodriguez, Kyung G. Cho, Kyu S. Lee, Charles B. Wilson, Michael S. B. Edwards, Victor A. Levin and Richard L. Davis

✓ The proliferative potential of low-grade astrocytomas was estimated in 47 patients. Each patient received an intravenous infusion of bromodeoxyuridine (BUdR), 150 to 200 mg/sq m, at the time of craniotomy to label cells in deoxyribonucleic acid (DNA) synthesis; the percentage of S-phase cells, or BUdR labeling index (LI), of each tumor was determined immunohistochemically. In 29 patients (60%), the tumors had BUdR LI's of less than 1%, indicating a slow growth rate; only three (10%) of these patients died of recurrent tumor during a follow-up period of up to 3½ years. In contrast, of the 18 patients (40%) whose tumors had BUdR LI's of 1% or more, 12 (67%) had a recurrence and nine died during the same follow-up period. These results show that the proliferative potential, as reflected by the BUdR LI, is an important prognostic factor that separates low-grade astrocytomas into two groups and provides a more scientific rationale for selecting treatment for individual patients.