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  • Author or Editor: Arthur J. DiPatri Jr x
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Benson P. Yang, Stefan A. Mindea and Arthur J. DiPatri Jr.

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Brian A. O'Shaughnessy, Arthur J. DiPatri Jr., Richard J. Parkinson and H. Hunt Batjer

✓The authors report the unique case of a 6-year-old African-American girl with sickle cell disease (SCD) and an associated moyamoya arteriopathy who developed a de novo arteriovenous malformation (AVM) of the cerebral circulation. Based on preoperative cerebral angiography, computerized tomography angiography, and magnetic resonance imaging, the incidentally discovered lesion was originally thought to be a direct arteriovenous fistula with an associated varix. At surgery, however, a 1.5-cm AVM was identified adjacent to the deep surface of the varix, and it was successfully resected. The diagnosis of cerebral AVM was then confirmed histopathologically. Based on a review of the literature, no published correlation between cerebral AVMs and SCD exists. In addition to reporting this case, the authors provide a description of AVM pathogenesis, with particular emphasis on acquired AVMs of the cerebral circulation.

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Brian A. O'Shaughnessy, Arthur J. DiPatri Jr., Richard J. Parkinson and H. Hunt Batjer

✓The authors report the unique case of a 6-year-old African-American girl with sickle cell disease (SCD) and an associated moyamoya arteriopathy who developed a de novo arteriovenous malformation (AVM) of the cerebral circulation. Based on preoperative cerebral angiography, computerized tomography angiography, and magnetic resonance imaging, the incidentally discovered lesion was originally thought to be a direct arteriovenous fistula with an associated varix. At surgery, however, a 1.5-cm AVM was identified adjacent to the deep surface of the varix, and it was successfully resected. The diagnosis of cerebral AVM was then confirmed histopathologically. Based on a review of the literature, no published correlation between cerebral AVMs and SCD exists. In addition to reporting this case, the authors provide a description of AVM pathogenesis, with particular emphasis on acquired AVMs of the cerebral circulation.

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Brian A. O'Shaughnessy, Kristian T. Schafernak, Arthur J. DiPatri Jr., Stewart Goldman and Tadanori Tomita

✓ Microfibrillar collagen hemostat (MCH), also known by its trade name Avitene, is commonly used to control hemorrhage during neurosurgery. Among the documented complications associated with this agent, a granulomatous foreign body reaction is rare, having been described in the central nervous system in only one previous clinical report. In the present study, the authors report the case of a 3-year-old boy who presented with a lesion which appeared to be the recurrence of a tumor 2 months after he had undergone gross-total resection for a medulloblastoma. The patient underwent resection of the presumed recurrent tumor, but histopathological analysis of the specimen revealed a granulomatous foreign body reaction to MCH and no tumor recurrence. In addition to describing the case, the authors review the surgical literature on foreign body reactions to MCH.

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Hideki Ogiwara, Douglas R. Nordli Jr., Arthur J. DiPatri Jr., Tord D. Alden, Robin M. Bowman and Tadanori Tomita

Object

Ganglioglioma is the most common neoplasm causing focal epilepsy, accounting for approximately 40% of all epileptogenic tumors and for 1–4% of all pediatric CNS tumors. The optimal surgical treatment for pediatric epileptogenic ganglioglioma has not been fully established. The authors present their experience in the surgical management of these lesions.

Methods

The authors retrospectively analyzed seizure outcome and surgical results of pediatric patients with ganglioglioma treated with resection. The patients' charts were reviewed for demographic data, clinical presentation, surgical therapy, and follow-up.

Results

The 30 patients (17 boys, 13 girls) had a history of medically intractable epilepsy. Total resection of tumor was achieved with or without adjacent epileptogenic tissue resection in all patients except 1, who underwent cyst fenestration and biopsy. Intraoperative electrocorticography (ECoG) was used in 21 patients. If an active spike focus or profound attenuation was observed in adjacent tissues, resection of those tissues was performed in addition to complete tumor resection (lesionectomy). The interval between onset of seizures and surgery ranged from 1 month to 9 years (mean 1.6 years). Patient age at the time of surgery ranged from 9 months to 16.3 years (mean 8.6 years). Twenty-five patients (83.3%) had complex partial seizures and 5 (16.7%) had simple partial seizures. Eighteen tumors (60%) were temporal (14 temporomesial, 4 temporolateral), and 12 (40%) were extratemporal. The mean follow-up period was 3.4 years (range 1 month–8.16 years). In 2 cases (6.7%), tumor recurrence was observed. Outcome was Engel Class I in 27 cases (90.0%; 14 temporomesial, 4 temporolateral, 9 extratemporal) and Engel Class II in 3 (10.0%; all extratemporal). Tumor resection allowed good seizure control, especially in the 18 cases of temporal ganglioglioma (all Engel Class I postoperatively). Eleven patients underwent removal of extratumoral epileptogenic tissue (anterior temporal neocortex resection in 10, anterior temporal neocortex resection with anterior hippocampectomy in 1) in addition to lesionectomy using intraoperative ECoG.

Conclusions

Lesionectomy with adjacent temporal neocortex resection using intraoperative ECoG provided good seizure control of pediatric temporal ganglioglioma. For extratemporal ganglioglioma, lesionectomy alone can provide fairly good seizure control. Considering the memory function of the hippocampus, lesionectomy with adjacent temporal neocortical resection can be a safe, feasible, and effective treatment option for epileptogenic gangliogliomas in pediatric patients.

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Maryam Soltanolkotabi, Samantha E. Schoeneman, Tord D. Alden, Michael C. Hurley, Sameer A. Ansari, Arthur J. DiPatri Jr., Tadanori Tomita and Ali Shaibani

Object

The authors undertook this study to assess the safety and efficacy of Onyx embolization in the treatment of intracranial arteriovenous malformations (AVMs) in pediatric patients.

Methods

All pediatric Onyx embolization of intracranial AVM cases performed consecutively at a single children's hospital over a 5-year period were collected and evaluated.

Results

Twenty-five patients (mean age 10.5 years) underwent a total of 38 procedures. An aggregate of 56 pedicles were embolized (mean 1.47 per session). The Spetzler-Martin grade was determined in all cases. Onyx embolization resulted in complete obliteration of the AVM in 3 cases (12%) and partial obliteration in 22 cases (88%). A total of 23 patients underwent surgical treatment. The mean preoperative AVM devascularization in these cases was 72%. One patient was treated with radiosurgery following Onyx embolization. Overall, 10 complications occurred in a total of 38 procedures (26.3%). None of the complications resulted in permanent neurological morbidity. The rate of transient neurological complications was 10.5% (4 of 38 procedures) and the rate of transient nonneurological complications was 5.3% (2 of 38 procedures). The remaining 4 complications were clinically silent (rate of 10.5%). There were no procedure-related deaths in this study population. There was no significant difference in patients with and without complications in terms of demographic characteristics, AVM grade, or embolization features (p ≥ 0.2). Deep venous drainage was associated with higher complication rates (p = 0.03).

Conclusions

Onyx utilization is feasible for preoperative or primary embolization in the treatment of pediatric intracranial AVMs; however, the spectrum of complications encountered is broad, and attention must be paid to the technical nuances of and indications for its use to avoid many potential dangerous effects. Although the overall complication rates were higher than expected, all were either clinically silent or had only transient clinical effects. Thus, this experience suggests that Onyx embolization can be performed safely with a low rate of permanent morbidity in pediatric patients harboring these difficult lesions.

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Sunit Das, Kenji Muro, Stewart Goldman, Veena Rajaram and Arthur J. DiPatri Jr.

✓ Teratomas, a group of nongerminomatous germ cell tumors, account for 3% of all childhood tumors. These unusual lesions reproduce the cellular and structural phenotypic traits associated with the three classic germ layers in inappropriate places in the body and often retain an embryonal character. These immature cells can differentiate into more malignant neoplasms. An intracranial location for this lesion type is rare. The authors describe the case of a 2-year-old boy with a teratoma of the posterior fossa that had partially differentiated into a medulloblastoma.

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Edward S. Ahn, Lawrence S. Chin, Kymberly A. Gyure, Richard S. Hudes, John Ragheb and Arthur J. DiPatri Jr.

✓ Clear cell meningioma (CCM) is a rare variant of meningioma characterized by sheets of polygonal cells with clear cytoplasm, a feature attributable to its high glycogen content. Authors have described its propensity to recur and metastasize despite its benign pathological characteristics. Clinical response to radiation in these reports has varied.

The authors present the case of a 7-year-old girl with a large petroclival CCM who underwent a staged subtotal resection and subsequent gamma knife surgery (GKS). Initially, the residual tumor decreased in size, but 6 years later, it had regrown (9 mm in size). A second GKS treatment was performed and the mass completely regressed without further complication.

The findings in this case suggest that GKS is a safe and effective adjunct for residual and recurrent CCM after resection. The delayed recurrence also emphasizes the importance of undertaking close follow-up examination after treating this potentially aggressive variant of meningioma.

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Daphne Li, Daniel M. Heiferman, Hasan R. Syed, João Gustavo Santos, Robin M. Bowman, Arthur J. DiPatri Jr., Tadanori Tomita, Nitin R. Wadhwani and Tord D. Alden

Atypical teratoid rhabdoid tumors (ATRTs) are rare malignant central nervous system tumors, commonly occurring before 3 years of age. Median overall survival (OS) of patients with these tumors is about 1 year, despite aggressive multimodal therapy. Pediatric primary spinal ATRTs are even more rare, with fewer than 50 cases reported. The authors present a series of four patients who were treated at Ann and Robert H. Lurie Children’s Hospital of Chicago in the period from 1996 to 2017.

These patients, with ages 2–11 years, presented with pain and a decline in motor functions. They were found to have lesions in the lumbar, thoracic, and/or cervical spine. One patient’s tumor was intramedullary with exophytic components, while another patient’s tumor had both intra- and extradural components. All patients underwent resection followed by chemotherapy (systemic and intrathecal). Two patients had fractionated radiation therapy and one had an autologous stem cell transplant. Three patients are known to be deceased (OS 8.5–45 months). The fourth patient was in remission 19 years after her initial diagnosis. To the authors’ knowledge, this is the largest series of pediatric primary spinal ATRTs documented at a single institution. These cases illustrate a variety of presentations of spinal ATRT and add to the body of literature on this aggressive pathology.

A systematic MEDLINE search was also conducted using the keywords “atypical teratoid rhabdoid tumor,” “pediatric spinal rhabdoid tumor,” and “malignant rhabdoid tumor spine.” Reports were included for patients younger than 21 years, without evidence of intracranial or systemic disease at the time of diagnosis. Clinical characteristics and outcomes of the four institutional cases were compared to those in the literature. This review yielded an additional 48 cases of primary pediatric spinal ATRTs reported in the English-language literature. Patients (ages 2 months to 19 years) presented with symptoms of pain, regression of motor function, and spinal cord compression. The majority of tumors were intradural (14 extramedullary, 8 intramedullary, 1 both). Eleven cases in the literature described tumors limited to extradural structures, while 10 tumors involved the intra- and extradural spine. Four reports did not specify tumor location. Although rare, spinal ATRT should be considered in the differential diagnosis of pediatric patients presenting with a new spinal mass.