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Gabriel Zada, Ning Lin and Edward R. Laws Jr.

Object

Growth patterns of pituitary adenomas have been observed to vary by histopathological subtype. The authors aimed to analyze variations in the patterns of extrasellar extension of nonfunctional macroadenomas (NFMAs) and growth hormone (GH)–secreting macroadenomas.

Methods

A retrospective review was conducted of data obtained in 75 patients who underwent transsphenoidal operations for histologically confirmed NFMAs (50 patients) and GH-secreting macroadenomas (25 patients) at the Brigham and Women's Hospital over an 18-month period. Patients with microadenomas and prior operations were excluded from the analysis. Preoperative MR images were reviewed to assess patterns of extrasellar extension in the varying tumor subtypes.

Results

The mean maximal tumor diameter in NFMAs and GH-secreting macroadenomas was 26 and 16 mm, respectively (p < 0.0001). Extension of the NFMAs occurred into the following regions: infrasellar, 23 patients (46%); suprasellar, 41 patients (82%); and cavernous sinus, 20 patients (40%). Extension of GH-macroadenomas occurred into the following regions: infrasellar, 18 patients (72%); suprasellar, 4 patients (16%); and cavernous sinus, 4 patients (16%). Compared with GH-adenomas, NFMAs were more likely to develop suprasellar extension (82% vs 16%, p < 0.0001), cavernous sinus extension (40% vs 16%, p = 0.04), and isolated suprasellar extension (30% vs 4%, p = 0.0145). GH-macroadenomas had higher overall rates of infrasellar extension (72% vs 46%, p < 0.05), and isolated infrasellar extension (52% vs 6%, p < 0.0001). Of the 13 GH-macroadenomas with isolated infrasellar extension, 5 (42%) met WHO criteria for atypical adenomas.

Conclusions

Substantial differences in extrasellar growth patterns were observed among varying histological subtypes of pituitary macroadenomas. Despite smaller tumor diameters, GH-macroadenomas demonstrated a proclivity for infrasellar extension, whereas NFMAs exhibited preferential extension into the suprasellar region.

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Giuseppe Lanzino

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Ning Lin, Clemens M. Schirmer and Mark R. Proctor

Disc cysts are rare intraspinal extradural lesions that communicate with the intervertebral disc and can mimic the symptoms of acute lumbar disc herniation. Initially reported in the Japanese-language literature as a new entity (discal cyst), there are few documented cases in North America, and only 1 prior case in the pediatric population. The authors present the case of a 16-year-old girl with an intervertebral disc cyst causing lumbar radiculopathy that progressed despite conservative treatment. All medical records, imaging studies, intraoperative findings, and pertinent literature were reviewed. Serial preoperative MR imaging revealed enlargement of the intraspinal cyst at the L4–5 level, resulting in compression of the right L-5 nerve root. Enlargement of the cyst occurred over a 4-month period despite conservative treatment with physical therapy and corticosteroid injections. Microsurgical discectomy and excision of the cyst resulted in complete resolution of the preoperative radiculopathy. An intervertebral disc cyst is a rare entity in the adult population and exceedingly rare in the pediatric population but should remain in the differential diagnosis of any intraspinal extradural mass. The authors hypothesize that there exists a spectrum of this entity that may not be responsive to conservative therapy. Cyst excision alone or in conjunction with microsurgical discectomy is safe and effective in treating radiculopathy caused by disc cysts.

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I-Duo Wang and Dueng-Yuan Hueng

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Bradley A. Gross, Ning Lin, Rose Du and Arthur L. Day

Literature reports on the natural history of cerebral cavernous malformations (CMs) are numerous, with considerable variability in lesion epidemiology, hemorrhage rates, and risk factors for hemorrhage. In this review, the authors performed a meta-analysis of 11 natural history studies. The overall male-to-female ratio was 1:1, and the mean age at presentation was 30.6 years. Overall, 37% of patients presented with seizures, 36% with hemorrhage, 23% with headaches, 22% with focal neurological deficits, and 10% were asymptomatic. Some patients had more than one symptom. Seizure presentation was most prevalent among supratentorial CMs, while focal neurological deficits were common in patients with infratentorial CMs. By location, CMs were in the cerebral hemispheres (66%), brainstem (18%), basal ganglia or thalamus (8%), cerebellum (6%), and other (2.5% [combined supra- and infratentorial, callosal or insular]). Overall, 19% of patients harbored multiple intracranial CMs, and 9% had radiographically apparent associated developmental venous anomalies. An overall annual hemorrhage rate of 2.4% per patient-year (range 1.6%–3.1%) was identified across 3 studies. Prior hemorrhage and female sex were risk factors for bleeding, while CM size and multiplicity did not affect hemorrhage rates. Although not impacting the hemorrhage rate itself, deep location was a risk factor for increased clinical aggressiveness.

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Dueng-Yuan Hueng and Yu-Chin An

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Ning Lin, Edward R. Smith, R. Michael Scott and Darren B. Orbach

OBJECT

The safe treatment of children using catheter-based angiography and embolization poses unique challenges because of the technical factors regarding the size and fragility of access and target vessels, as well as unique pediatric cerebrovascular pathologies. The complication rates for neurointerventional procedures in children have not been established.

METHODS

The records of a consecutive cohort of pediatric patients who underwent neuroangiography and/or embolization between 2007 and 2013 were reviewed retrospectively to identify both intraprocedural and postprocedural complications. Demographic and clinical risk factors were analyzed with a multivariate logistic regression model.

RESULTS

The 697 consecutive procedures consisted of 429 diagnostic angiograms and 268 embolizations (mean age of patients 11.1 years; range 4 days to 18 years; 217 females). There were 130 intracranial, 122 extracranial, and 16 spinal embolizations. Pathologies included 28 intracranial arteriovenous malformations (AVMs), 12 spinal AVMs, 19 aneurysms, 29 vein of Galen malformations, 29 dural arteriovenous fistulas, 96 extracranial AVMs, 39 tumors, 3 strokes, and 13 others. Overall, 2 intraprocedural and 1 postprocedural complication (0.7%) occurred in the diagnostic group, all of which were nonneurological events. In the embolization group, 7 intraprocedural and 11 postprocedural complications (6.7%) were observed. Of these complications, 15 were nonneurological events (5.6%), 1 was a short-term neurological event (0.4%), and 2 were long-term neurological events (0.7%).

CONCLUSIONS

Neither the technical challenges posed by children’s access and target vessels nor the unique neuro-vascular pathologies seen in children need result in an elevated morbidity rate related to neuroangiography and embolization. At a dedicated high-volume center, the complication rates may be lower than those for comparable procedures performed in adults.

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Amit Singla, Ning Lin, Allen L. Ho, R. Michael Scott and Edward R. Smith

Surgically created openings such as bur holes can serve as avenues for the development of collateral blood supply to the brain in patients with moyamoya disease. When such collateralization occurs through preexisting shunt catheter sites, the potential exists for perioperative stroke if these vessels are damaged during revision of a ventricular catheter for shunt malfunction. In this paper the authors report on a series of patients with a history of ventriculoperitoneal (VP) shunts who later developed moyamoya disease and were found to have spontaneous transdural collateral vessels at ventricular catheter sites readily visualized on diagnostic angiography. A consecutive surgical series of 412 patients with moyamoya disease treated at Boston Children's Hospital from 1990 to 2010 were reviewed to identify patients with concomitant moyamoya and a VP shunt. The clinical records and angiograms of these patients were reviewed to determine the extent of bur hole collaterals through the shunt site. Three patients were identified who had VP shunts placed for hydrocephalus and subsequently developed moyamoya disease. All 3 patients demonstrated spontaneous transdural collaterals at the ventricular catheter bur hole, as confirmed by angiography during the workup for moyamoya disease. No patients required subsequent revision of their ventricular catheters following the diagnosis of moyamoya. All patients have remained stroke free and clinically stable following pial synangiosis. Although the association of moyamoya and shunted hydrocephalus is rare, it may present a significant potential problem for the neurosurgeon treating a shunt malfunction in this patient population, because shunt bur holes may become entry sites for the ingrowth of significant cortical transdural collateral blood supply to the underlying brain. Shunt revision might therefore be associated with an increased risk of postoperative stroke or operative-site hemorrhage in this population if this vascularization is interrupted when shunt catheters are removed and replaced. A knowledge of the existence of shunt-related collaterals in patients with moyamoya may aid the surgeon in planning shunt revisions and considering, for example, a new entry point for a ventricular catheter, rather than replacing an existing one, to minimize the risk of jeopardizing existing collaterals.

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Eric M. Jackson, Ning Lin, Sunil Manjila, R. Michael Scott and Edward R. Smith

Object

Patients with moyamoya who are younger than 2 years of age represent a therapeutic challenge because of their frequent neurological instability and concomitant anesthetic risks. The authors report their experience with pial synangiosis revascularization in this population.

Methods

The authors reviewed the clinical and radiographic records of all patients with moyamoya in a consecutive series of patients under 2 years of age, who underwent cerebral revascularization surgery using pial synangiosis at a single institution.

Results

During a 12-year period (1994–2005), 34 procedures (bilateral in 15 patients, unilateral in 4) were performed in 19 patients younger than 2 years (out of a total of 456 procedures in 240 patients). Eighteen of these patients presented with either stroke or transient ischemic attack. The average age of the 19 patients at first surgery was 1.4 years (range 6 months–1.9 years). Unanticipated staged operations occurred in 3 patients, due to persistent electroencephalographic changes during the initial surgery in 2 cases and due to brain swelling during the procedure requiring ventriculostomy in the other. There were 2 perioperative strokes; both patients had postoperative seizures but made clinical recoveries. The average follow-up was 7 years (range 1–14 years). Long term, at follow-up, 13 patients (68%) were clinically independent for their age, with 8 (42%) having no significant deficit. Late complications included subdural hygroma evacuation (1), additional revascularization procedures performed years later for frontal lobe ischemia (2), late infarction (1), and asymptomatic ischemic change on routine follow-up MRI studies (1). All patients who had both pre- and postoperative angiography demonstrated progression of disease.

Conclusions

Despite the challenges inherent to this population, the majority of children with moyamoya under the age of 2 years have a good long-term prognosis. The data from this study support the use of pial synangiosis as a safe, effective, and durable method for treatment of moyamoya for most children in this potentially high-risk population.

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Andrew Shaw and E. Antonio Chiocca