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Luca Massimi, Giuseppe Maria Della Pepa, Gianpiero Tamburrini, and Concezio Di Rocco

Chiari malformation Type I (CM-I) is usually suspected in patients with slowly progressing neurological symptoms. However, in some instances, especially if syringomyelia is associated, an abrupt clinical onset is reported and is accompanied by an acknowledged risk of potentially severe clinical signs or even sudden death. Little is known about such a critical course in CM-I/syringomyelia complex.

The authors describe 3 challenging cases of the abrupt onset of CM-I/syringomyelia to reveal more information on the clinical presentation and pathogenetic mechanisms of this sudden and potentially severe clinical phenomenon: a 38-year-old man experienced acute respiratory failure requiring intubation following acute decompensation of hydrocephalus associated with Noonan syndrome, a 1-year-old boy had sudden hemiparesis and Horner syndrome after a minor head/neck injury, and a 2.5-year-old boy presented with quickly progressing tetraplegia and dyspnea after a mild flexion and extension neck injury a few hours before. All 3 patients showed a CM-I/syringomyelia complex at diagnosis, and all of them had a good neurological outcome after surgery despite the ominous clinical presentation.

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Gianpaolo Petrella, Gianpiero Tamburrini, Libero Lauriola, and Concezio Di Rocco

✓Spinal angiolipomas are rare, benign lesions representing 0.14 to 1.2% of all spinal axis tumors. They most commonly involve the midthoracic spine and are located in the posterior epidural space. Up to now, six pediatric cases have been reported in the literature; two of them involved an acute clinical onset that was related to a venous infarction of a tumor. The authors report the case of a 16-year-old boy with a midthoracic epidural angiolipoma who was admitted with a clinical history of an acute paraparesis. In contrast to previous descriptions, the acute onset in this case was related to a spontaneous intratumoral abscess within the tumor.

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Vera Vigo, Domenica Immacolata Battaglia, Paolo Frassanito, Gianpiero Tamburrini, Massimo Caldarelli, and Luca Massimi

Cephalohematoma, one of the most common neonatal head injuries, generally undergoes spontaneous resorption. When calcified, it may cause cranial vault distortion and depression of the inner skull layer, although it remains asymptomatic. Surgery, indeed, is usually performed for cosmetic purposes. For these reasons, the long-term effects of calcified cephalohematoma (CC) are widely unknown.

The authors report the case of an 11-year-old girl with a persistent calcified CC causing skull deformity and delayed electroencephalography (EEG) anomalies. These anomalies were detected during routine control EEG and were not clinically evident. The young girl underwent surgical removal of the CC for cosmetic purpose. The EEG abnormalities disappeared after surgery, thus reinforcing the hypothesis of a correlation with the brain “compression” resulting from the CC. To the best of the authors' knowledge this is the first time that CC-associated EEG anomalies have been described: even though these anomalies cannot be considered an indication for surgery, they merit late follow-up in case of skull deformity.

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Paolo Frassanito, Georgios Markogiannakis, Rina Di Bonaventura, Luca Massimi, Gianpiero Tamburrini, and Massimo Caldarelli

Descending transtentorial herniation (DTH) is a complication of raised pressure in the supratentorial compartment, usually resulting from mass lesion of several etiologies. The authors report an exceptional case of DTH complicating the implant of a CSF shunting device in the trapped fourth ventricle of a 17-year-old boy in whom a second CSF shunting device had been implanted for neonatal posthemorrhagic and postinfectious hydrocephalus. The insidious clinical and radiological presentation of DTH, mimicking a malfunction of the supratentorial shunt, is documented. Ultimately, the treatment consisted of removal of the infratentorial shunt and endoscopic acqueductoplasty with stenting. The absence of supratentorial mass lesion and other described etiologies of DTH prompted the authors to speculate on the hydrodynamic pathogenesis of DTH in the present case.

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Benedetta Ludovica Pettorini, Paolo Frassanito, Massimo Caldarelli, Gianpiero Tamburrini, Luca Massimi, and Concezio Di Rocco

Craniopharyngioma has long been considered a benign tumor because of its pathological aspect. This primordial view of craniopharyngioma fit with the primitive treatment attempts based on blind resection of the tumor each time it recurred. The limits of this management strategy were proven early by the high morbidity related to the resection and recurrence risk despite radical lesion removal. Nowadays, craniopharyngioma must be considered a complex molecular disease, and a detailed explanation of the mechanisms underlying its aggressive biological and clinical behavior, despite some benign pathological features, would be the first step toward defining the best management of craniopharyngioma. Indeed, advances in the knowledge of the molecular mechanisms at the base of craniopharyngioma oncogenesis will lead to comprehension of the critical checkpoints involved in neoplastic transformation. The final research target will be the definition of new biological agents able to reverse the neoplastic process by acting on these critical checkpoints. This biological approach will lead to a refined therapy combining higher efficacy and safety with lower morbidity. In this paper the authors reveal state-of-the-art comprehension of the molecular biology of craniopharyngioma and the consequent therapeutic implications.

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Massimo Caldarelli, Federica Novegno, Luca Massimi, Rossana Romani, Gianpiero Tamburrini, and Concezio Di Rocco

Object

Despite progress in the understanding of the causes and pathophysiology of Chiari malformation Type I (CM-I), definitive surgical treatment remains unresolved. Various techniques have been propounded over the years, but there is no general consensus on the most appropriate surgical management for this condition. The authors report their experience with the surgical treatment of 30 pediatric patients with CM-I.

Methods

The results obtained in 30 patients who underwent surgery for symptomatic CM-I with a purely extradural procedure were retrospectively reviewed. The patient age at operation ranged from 2 months to 16 years (mean 68 months). In 26 patients the CM-I occurred as an isolated disease, whereas in four a recognized bone anomaly involving the cranial base (achondroplasia) was observed. All patients underwent suboccipital craniectomy, which was accompanied by C-l laminectomy in 22. In all cases a thick fibrous band at the level of foramen magnum was resected; in 11 children serial incisions of the outer layer of the dura mater were performed as well to expand the posterior fossa volume. The follow-up duration varied from a minimum of 12 months to 12.6 years (mean 4.7 years).

The most frequent symptoms and signs were head and/or neck pain (56.7%), followed by vertigo (27.7%), upper-and lower-extremity weakness (20.0%), and ataxia (20.0%). Syringomyelia was noted in 12 patients (40.0%) on magnetic resonance (MR) images. Improvement in or resolution of clinical symptoms and signs was observed in all patients. However, only minimal or no modifications could be found in the position of cerebellar tonsils on postoperative MR images in most patients (only occasional improvement in tonsillar herniation), whereas syringomyelic cavitations reduced in size in half of the cases. Nevertheless, in only two patients was a reoperation necessary.

Conclusions

In the authors' experience, suboccipital craniectomy and C-l laminectomy (eventually integrated by dural delamination) can represent an effective treatment for symptoms associated with CM-I.

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Daniela Chieffo, Gianpiero Tamburrini, Massimo Caldarelli, and Concezio Di Rocco

Object

Functional involvement of the thalamus in cognitive processing has been only anecdotally reported in the literature, and these cases are mostly related to thalamic hemorrhages; there is no available information on cognitive development in children with thalamic tumors.

Methods

All children admitted with a diagnosis of thalamic tumor at the authors' institution between January 2008 and January 2011 were considered for the present study. Exclusion criteria were age less than 18 months and the presence of severe neurological deficits, both of which prevented a reliable neuropsychological evaluation. A complete preoperative neuropsychological evaluation was performed.

Results

Twenty children were selected (mean age 102.4 months). Total IQ was in the normal range in all patients (mean 90.1, SD 13.87) with a significant difference between verbal IQ (mean 97.70, SD 17.77) and performance IQ (mean 84.82, SD 17.01). A significant correlation was found between global cognitive impairment and a histological finding of low-grade tumors (p < 0.001). Children with a mesial thalamic tumor had a higher working memory deficit and delayed recall disorders (p < 0.001). Naming disorders were related to the presence of a bilateral (p < 0.001) or mesial (p < 0.001) thalamic tumor, without a significant difference between left or right hemisphere involvement. A significant correlation was also found between the presence of neurolinguistic disorders and mesially located tumors (p < 0.001). Children with right-sided tumors more frequently had constructional apraxia and executive function disorders (p < 0.001).

Conclusions

The present study suggests that thalamic tumors in different locations might have specific neuropsychological profiles.

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Benedetta L. Pettorini, Gianpiero Tamburrini, Luca Massimi, Massimo Caldarelli, and Concezio Di Rocco

The intracystic injection of chemo- and radiotherapeutic agents was introduced for the treatment of craniopharyngioma to control tumor growth and to delay the potentially harmful effects of surgery or radiation therapy. The positioning of cyst catheters has been performed by means of direct vision, stereotactically guided insertion, and ultrasonographic and ventriculoscopic guidance. The insertion of a catheter into the cyst is not devoid of complications, with an incidence ranging up to 16%, independent of the surgical technique used.

Eight patients (mean age 25.8 years) with symptomatic cystic craniopharyngioma were treated by means of an endoscopic transventricular approach for the insertion of an intracystic catheter for intratumoral therapy with interferon-α.

A single right precoronal bur hole is made, and the frontal horn of the lateral ventricle is accessed under neuronavigation guidance. A ventricular catheter with an inserted stylet was advanced anterior to the endoscope sheath through the same cortical access as the endoscope and was guided under endoscopic view down to the cyst dome wall. The coagulated surface of the craniopharyngioma cyst was punctured and the tip of the ventricular catheter was advanced; the depth was established preoperatively on MR scans and confirmed by neuronavigation guidance. The proximal end of the cystic catheter was connected to an access chamber to be left in the subcutaneous space, and the endoscope was slowly retracted.

The authors' experience favors the use of neuroendoscopic positioning of intracystic catheters as safer than open and stereotactic approaches.

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Benedetta Ludovica Pettorini, Paolo Frassanito, Gianpiero Tamburrini, Luca Massimi, Massimo Caldarelli, and Concezio Di Rocco

Multilocular hydrocephalus usually requires placement of multiple ventricular catheters for the treatment of secondary cysts and intraventricular septation. The formation of strong adhesions can embed the catheters so that they cannot be removed without a higher risk of intraventricular hemorrhage. Moreover, the devices could represent a nidus for infection and a risk for formation and enlargement of intraventricular secondary cysts. Neuroendoscopy allows the surgeon safely to reach the ventricular catheters that have been left or lost in the ventricular cavity, and to remove them via a minimally invasive approach. In this paper, the authors document another application of ventricular endoscopy in the management of this kind of hydrocephalus. The removal of an unnecessary and no longer useful prosthetic device that is a possible focus of infection justifies endoscopic treatment, which, in this experience, is not associated with morbidity.