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Dimitrios C. Nikas, Alexander F. Post, Asim F. Choudhri, Catherine A. Mazzola, Laura Mitchell and Ann Marie Flannery

Object

The objective of this systematic review is to answer the following question: Does ventricle size after treatment have a predictive value in determining the effectiveness of surgical intervention in pediatric hydrocephalus?

Methods

The US National Library of Medicine PubMed/MEDLINE database and the Cochrane Database of Systematic Reviews were searched using MeSH headings and key words relevant to change in ventricle size after surgical intervention for hydrocephalus in children. An evidentiary table was assembled summarizing the studies and the quality of evidence (Classes I–III).

Results

Six articles satisfied inclusion criteria for the evidentiary tables for this part of the guidelines. All were Class III retrospective studies.

Conclusions

Recommendation: There is insufficient evidence to recommend a specific change in ventricle size as a measurement of the effective treatment of hydrocephalus and as a measurement of the timing and effectiveness of treatments including ventriculoperitoneal shunts and third ventriculostomies. Strength of Recommendation: Level III, unclear clinical certainty.

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Paul Klimo Jr., Cody L. Nesvick, Alberto Broniscer, Brent A. Orr and Asim F. Choudhri

OBJECT

Malignant tumors of the brainstem, excluding classic diffuse intrinsic pontine gliomas (DIPGs), are a very rare, heterogeneous group of neoplasms that have been infrequently described in the literature. In this paper, the authors present their experiences with treating these unique cancers.

METHODS

A retrospective chart review was conducted to identify eligible cases over a 15-year period. All tumors involving the pons were, by consensus, felt not to be DIPGs based on their neuroimaging features. Demographic information, pathological specimens, neuroimaging characteristics, surgical and nonsurgical management plans, and survival data were gathered for analysis.

RESULTS

Between January 2000 and December 2014, 29 patients were identified. The mean age at diagnosis was 8.4 years (range 2 months to 25 years), and 17 (59%) patients were male. The most common presenting signs and symptoms were cranial neuropathies (n = 24; 83%), hemiparesis (n = 12; 41%), and ataxia or gait disturbance (n = 10; 34%). There were 18 glial and 11 embryonal tumors. Of the glial tumors, 5 were radiation-induced and 1 was a malignant transformation of a previously known low-grade tumor. Surgical intervention consisted of biopsy alone in 12 patients and some degree of resection in another 15 patients. Two tumors were diagnosed postmortem. The median overall survival for all patients was 196 days (range 15 to 3999 days). There are currently 5 (17%) patients who are still alive: 1 with an anaplastic astrocytoma and the remaining with embryonal tumors.

CONCLUSIONS

In general, malignant non-DIPG tumors of the brainstem carry a poor prognosis. However, maximal cytoreductive surgery may be an option for select patients with focal tumors. Long-term survival is possible in patients with nonmetastatic embryonal tumors after multimodal treatment, most importantly maximal resection.

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Nickalus R. Khan, Kenneth Moore, Jaafar Basma, David S. Hersh, Asim F. Choudhri, Brandy Vaughn and Paul Klimo Jr.

OBJECTIVE

An ischemic stroke following an elective craniotomy in a child is perceived to be a rare event. However, to date there are few papers on this topic. The purpose of this study was to investigate the occurrence of stroke following elective intracranial surgery at a children’s hospital.

METHODS

The authors performed a retrospective review of all patients who developed a perioperative stroke following an elective craniotomy from 2010 through 2017. Data were collected using an institutional database that contained demographic, medical, radiological, and outcome variables.

RESULTS

A total of 1591 elective craniotomies were performed at the authors’ institution during the study period. Of these, 28 (1.8%) were followed by a perioperative stroke. Radiographic diagnosis of the infarction occurred at a median of 1.7 days (range 0–9 days) from the time of surgery, and neurological deficits were apparent within 24 hours of surgery in 18 patients (62.5%). Infarcts tended to occur adjacent to tumor resection sites (86% of cases), and in a unilateral (89%), unifocal (93%), and supratentorial (93%) location. Overall, 11 (39.3%) strokes were due to a perforating artery, 10 (35.7%) were due to a large vessel, 4 (14.3%) were venous, and 3 (10.7%) were related to hypoperfusion or embolic causes. Intraoperative MRI (iMRI) was used in 11 of the 28 cases, and 6 (55%) infarcts were not detected, all of which were deep.

CONCLUSIONS

The incidence of stroke following an elective craniotomy is low, with nearly all cases (86%) occurring after tumor resection. Perforator infarcts were most common but may be missed on iMRI.

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Frederick A. Boop, Berkeley Bate, Asim F. Choudhri, Brian Burkholder and Paul Klimo Jr.

The development of high-quality intraoperative MRI (iMRI) capability has offered a major advance in the care of patients with complex intracranial disease. To date, this technology has been limited by the need for pin fixation of the calvaria. The authors report their preliminary experience with an MRI-compatible horseshoe headrest that allows for the following: 1) iMRI in patients too young for pin fixation; 2) iMRI in patients with large calvarial defects; 3) the ability to move the head during iMRI surgery; and 4) the use of neuronavigation in such cases. The authors report 2 cases of infants in whom the Visius Surgical Theatre horseshoe headrest (IMRIS Inc.) was used. Image quality was equivalent to that of pin fixation. The infants suffered no skin issues. The use of neuronavigation with the system remained accurate and could be updated with the new iMRI information. The Visius horseshoe headrest offers a technical advance in iMRI technology for infants, for patients with cranial defects or prior craniotomies in whom pin fixation may not be safe, or for patients in whom the need to move the head during surgery is required. The image quality of the system remains excellent, and the ability to merge new images to the neuronavigation system is helpful.

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Joseph H. McAbee, Joseph Modica, Clinton J. Thompson, Alberto Broniscer, Brent Orr, Asim F. Choudhri, Frederick A. Boop and Paul Klimo Jr.

OBJECT

Cervicomedullary tumors (CMTs) represent a heterogeneous group of intrinsic neoplasms that are typically low grade and generally carry a good prognosis. This single-institution study was undertaken to document the outcomes and current treatment philosophy for these challenging neoplasms.

METHODS

The charts of all pediatric patients with CMTs who received treatment at St. Jude Children’s Research Hospital between January 1988 and May 2013 were retrospectively reviewed. Demographic, surgical, clinical, radiological, pathological, and survival data were collected. Treatment-free survival and overall survival were estimated, and predictors of recurrence were analyzed.

RESULTS

Thirty-one children (16 boys, 15 girls) with at least 12 months of follow-up data were identified. The median age at diagnosis was 6 years (range 7 months-17 years) and the median follow-up was 4.3 years. Low-grade tumors (Grade I or II) were present in 26 (84%) patients. Thirty patients underwent either a biopsy alone or resection, with the majority of patients undergoing biopsy only (n = 12, 39%) or subtotal resection (n = 14, 45%). Only 4 patients were treated solely with resection; 21 patients received radiotherapy alone or in combination with other treatments. Recurrent tumor developed in 14 children (45%) and 4 died as a result of their malignancy. A high-grade pathological type was the only independent variable that predicted recurrence. The 5- and 10-year treatment-free survival estimates are 64.7% and 45.3%, respectively. The 5- and 10-year overall survival estimate is 86.7%.

CONCLUSIONS

Children with CMTs typically have low-grade neoplasms and consequently long-term survival, but high risk of recurrence. Therapy should be directed at achieving local tumor control while preserving and even restoring neurological function.

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David S. Hersh, Kenneth Moore, Vincent Nguyen, Lucas Elijovich, Asim F. Choudhri, Jorge A. Lee-Diaz, Raja B. Khan, Brandy Vaughn and Paul Klimo Jr.

OBJECTIVE

Stenoocclusive cerebral vasculopathy is an infrequent delayed complication of ionizing radiation. It has been well described with photon-based radiation therapy but less so following proton-beam radiotherapy. The authors report their recent institutional experience in evaluating and treating children with radiation-induced cerebral vasculopathy.

METHODS

Eligible patients were age 21 years or younger who had a history of cranial radiation and subsequently developed vascular narrowing detected by MR arteriography that was significant enough to warrant cerebral angiography, with or without ischemic symptoms. The study period was January 2011 to March 2019.

RESULTS

Thirty-one patients met the study inclusion criteria. Their median age was 12 years, and 18 (58%) were male. Proton-beam radiation therapy was used in 20 patients (64.5%) and photon-based radiation therapy was used in 11 patients (35.5%). Patients were most commonly referred for workup as a result of incidental findings on surveillance tumor imaging (n = 23; 74.2%). Proton-beam patients had a shorter median time from radiotherapy to catheter angiography (24.1 months [IQR 16.8–35.4 months]) than patients who underwent photon-based radiation therapy (48.2 months [IQR 26.6–61.1 months]; p = 0.04). Eighteen hemispheres were revascularized in 15 patients. One surgical patient suffered a contralateral hemispheric infarct 2 weeks after revascularization; no child treated medically (aspirin) has had a stroke to date. The median follow-up duration was 29.2 months (IQR 21.8–54.0 months) from the date of the first catheter angiogram to last clinic visit.

CONCLUSIONS

All children who receive cranial radiation therapy from any source, particularly if the parasellar region was involved and the child was young at the time of treatment, require close surveillance for the development of vasculopathy. A structured and detailed evaluation is necessary to determine optimal treatment.

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Catherine A. Mazzola, Asim F. Choudhri, Kurtis I. Auguste, David D. Limbrick Jr., Marta Rogido, Laura Mitchell and Ann Marie Flannery

Object

The objective of this systematic review and analysis was to answer the following question: What are the optimal treatment strategies for posthemorrhagic hydrocephalus (PHH) in premature infants?

Methods

Both the US National Library of Medicine and the Cochrane Database of Systematic Reviews were queried using MeSH headings and key words relevant to PHH. Two hundred thirteen abstracts were reviewed, after which 98 full-text publications that met inclusion criteria that had been determined a priori were selected and reviewed.

Results

Following a review process and an evidentiary analysis, 68 full-text articles were accepted for the evidentiary table and 30 publications were rejected. The evidentiary table was assembled linking recommendations to strength of evidence (Classes I–III).

Conclusions

There are 7 recommendations for the management of PHH in infants. Three recommendations reached Level I strength, which represents the highest degree of clinical certainty. There were two Level II and two Level III recommendations for the management of PHH.

Recommendation Concerning Surgical Temporizing Measures: I. Ventricular access devices (VADs), external ventricular drains (EVDs), ventriculosubgaleal (VSG) shunts, or lumbar punctures (LPs) are treatment options in the management of PHH. Clinical judgment is required. Strength of Recommendation: Level II, moderate degree of clinical certainty.

Recommendation Concerning Surgical Temporizing Measures: II. The evidence demonstrates that VSG shunts reduce the need for daily CSF aspiration compared with VADs. Strength of Recommendation: Level II, moderate degree of clinical certainty.

Recommendation Concerning Routine Use of Serial Lumbar Puncture: The routine use of serial lumbar puncture is not recommended to reduce the need for shunt placement or to avoid the progression of hydrocephalus in premature infants. Strength of Recommendation: Level I, high clinical certainty.

Recommendation Concerning Nonsurgical Temporizing Agents: I. Intraventricular thrombolytic agents including tissue plasminogen activator (tPA), urokinase, or streptokinase are not recommended as methods to reduce the need for shunt placement in premature infants with PHH. Strength of Recommendation: Level I, high clinical certainty.

Recommendation Concerning Nonsurgical Temporizing Agents. II. Acetazolamide and furosemide are not recommended as methods to reduce the need for shunt placement in premature infants with PHH. Strength of Recommendation: Level I, high clinical certainty.

Recommendation Concerning Timing of Shunt Placement: There is insufficient evidence to recommend a specific weight or CSF parameter to direct the timing of shunt placement in premature infants with PHH. Clinical judgment is required. Strength of Recommendation: Level III, unclear clinical certainty.

Recommendation Concerning Endoscopic Third Ventriculostomy: There is insufficient evidence to recommend the use of endoscopic third ventriculostomy (ETV) in premature infants with posthemorrhagic hydrocephalus. Strength of Recommendation: Level III, unclear clinical certainty.

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David S. Hersh, Rahul Kumar, Kenneth A. Moore, Luke G. F. Smith, Christopher L. Tinkle, Jason Chiang, Zoltan Patay, Amar Gajjar, Asim F. Choudhri, Jorge A. Lee-Diaz, Brandy Vaughn and Paul Klimo Jr.

OBJECTIVE

Biopsies of brainstem lesions are performed to establish a diagnosis in the setting of an atypical clinical or radiological presentation, or to facilitate molecular studies. A better understanding of the safety and diagnostic yield of brainstem biopsies would help guide appropriate patient selection.

METHODS

All patients who underwent biopsy of a brainstem lesion during the period from January 2011 to June 2019 were reviewed. Demographic, radiological, surgical, and outcome data were collected.

RESULTS

A total of 58 patients underwent 65 brainstem biopsies during the study period. Overall, the median age was 7.6 years (IQR 3.9–14.2 years). Twenty-two of the 65 biopsies (34%) were open, 42 (65%) were stereotactic, and 1 was endoscopic. In 3 cases (5%), a ventriculoperitoneal shunt was placed, and in 9 cases (14%), a posterior fossa decompression was performed during the same operative session as the biopsy. An intraoperative MRI (iMRI) was performed in 28 cases (43%). In 3 of these cases (11%), the biopsy was off target and additional samples were obtained during the same procedure. New neurological deficits were noted in 5 cases (8%), including sensory deficits, ophthalmoparesis/nystagmus, facial weakness, and hearing loss; these deficits persisted in 2 cases and were transient in 3 cases. A pseudomeningocele occurred in 1 patient; no patients developed a CSF leak or infection. In 8 cases (13%) an additional procedure was needed to obtain a diagnosis.

CONCLUSIONS

Brainstem biopsies are safe and effective. Target selection and approach should be a collaborative effort. iMRI can be used to assess biopsy accuracy in real time, thereby allowing any adjustment if necessary.

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Ann Marie Flannery, Catherine A. Mazzola, Paul Klimo Jr., Ann-Christine Duhaime, Lissa C. Baird, Mandeep S. Tamber, David D. Limbrick Jr., Dimitrios C. Nikas, Joanna Kemp, Alexander F. Post, Kurtis I. Auguste, Asim F. Choudhri, Laura S. Mitchell and Debby Buffa