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Federico Di Rocco, Masami Yoshino and Shizuo Oi

Object

Although in recent years endoscopic procedures have been used for intracranial arachnoid cysts with favorable preliminary results in certain locations, optimal surgical treatment is still controversial. The purpose of this study was to evaluate the efficacy and safety of endoscopic transventricular ventriculocystostomy in the treatment of intracranial cysts based on the concept of normalizing cerebrospinal fluid (CSF) dynamics.

Methods

Twelve symptomatic pediatric patients with congenital intracranial cysts underwent surgery at Jikei University in Tokyo. A neuroendoscopic transventricular ventriculocystostomy was performed in nine patients and an endoscope-assisted craniotomy in the remaining three. Endoscopy was performed using a freehand maneuver with a newly designed rigid-rod neuroendoscope that is frameless and has a small diameter.

Clinical results were good in all patients, although cysts in three were not prominently reduced in size when follow-up imaging studies were performed. Neither death nor symptomatic morbidity occurred, and no patient required shunt placement. In three cases the endoscopic fenestration was associated with an endoscopic third ventriculostomy (ETV). Postoperative CSF dynamics studies consisting of computerized tomography ventriculocysternography, and pre- and postoperative cine-mode magnetic resonance imaging demonstrated free communication between fenestrated cysts and ventricular/cistern CSF pathways consistent with normalization of CSF dynamics.

Conclusions

Neuroendoscopic transventricular ventriculocystostomy constitutes a valid alternative to microsurgery for intracranial cysts located within or adjacent to the ventricles. It creates an effective CSF flow within the cyst with minimal alteration of subarachnoid spaces. It may be combined with an ETV procedure in case of obstruction of CSF pathways and should be preferred to the insertion of shunts.

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Federico Di Rocco, Eric Arnaud and Dominique Renier

Object

The prevalence of the different subtypes of craniosynostosis varies greatly. The aim of this study was to analyze the prevalences of the different subtypes of craniosynostosis at a single major craniofacial center and their changes during a 20-year period.

Methods

The medical charts of 2808 children hospitalized between 1988 and 2007 for a true craniosynostosis were retrospectively reviewed. Patients were divided according to their subtype of craniosynostosis (sagittal, plagiocephaly, brachycephaly, metopic, oxycephaly, syndromic cases, or unclassifiable).

Results

The total number of patients according to their craniosynostosis subtype were as follows: 1224 cases of scaphocephaly, 315 cases of plagiocephaly, 598 cases of trigonocephaly, 105 cases of brachycephaly, 69 cases of oxycephaly, 396 syndromic cases, and 101 unclassifiable cases. The prevalences of these craniosynostoses evolved differently over time. The number of children hospitalized each year increased over time from 106 to 181 cases, but this increase varied according to the subtype of craniosynostosis. Cases of scaphocephaly, plagiocephaly, brachycephaly, and syndromic cases increased similarly (170% increase on average), whereas trigonocephalies dramatically increased (420% increase).

Conclusions

The evolution observed in the prevalence of craniosynostosis might result from several factors. An increased recruitment of patients at the center and an improvement in the diagnosis of craniosynostosis might explain the overall increase. However, other mechanisms should be examined to explain the great increase in the number of cases of trigonocephaly (environmental or pharmacological).

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Federico Di Rocco and Shizuo Oi

✓Hadju—Cheney syndrome (HCS) is a rare autosomal-dominant disorder with variable expressivity. It is characterized by facial dysmorphism, premature tooth loss, osteolysis of distal phalanges, and skull abnormalities. In some cases, progressive platybasia can occur and can lead to Chiari malformation with an obstruction of cerebrospinal fluid flow. To the best of the authors' knowledge, only five cases of HCS-associated syringomyelia have been reported in the literature. Because of the rarity of this association, little is known about its natural history.

The authors present the case of a 16-year-old boy affected by HCS. On initial magnetic resonance (MR) imaging, a severe basilar invagination with Chiari malformation and cervicothoracic syringomyelia was documented. The syringomyelia had no clinical manifestations. A repeated MR image demonstrated a spontaneous resolution of the syrinx with no changes in the tonsil or the platybasia. The regression of the syringomyelia was confirmed by a control MR imaging examination performed after a 2-year period. No changes in the patient's clinical conditions were found during the follow-up period. This is the first case of spontaneous regression of the syringomyelia despite a severe platybasia in HCS. It did not appear correlated to a modification of the tonsil's structure or position. This observation illustrates one possible evolution of syringomyelia in the natural history of HCS and raises the question of the potential mechanisms involved in the spontaneous drainage of the syringomyelic cavity.

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Tatiana Protzenko Cervante, Eric Arnaud, Francis Brunelle and Federico Di Rocco

OBJECTIVE

The sagittal suture is usually considered an external anatomical landmark, indicating the location of the superior sagittal sinus (SSS) for surgical approaches. Children with unilateral coronal synostosis (UCS) often present with an important deviation of the sagittal suture. Because these patients usually undergo frontal reconstruction or even endoscope-assisted minimally invasive procedures, it is imperative to know the location of the SSS. The aim of this investigation was to study the anatomical relationship between the SSS and the sagittal suture in children with anterior plagiocephaly.

METHODS

The authors retrospectively studied the relationship between the sagittal sinus and the sagittal suture at 5 points: nasion, midpoint nasion-bregma, bregma, midpoint bregma-lambda, and lambda. The study analyzed CT scans of 50 children with UCS admitted to the craniofacial unit of Necker Enfants Malades Hospital between March 2006 and March 2013 and compared them with 50 control children with no evidence of craniosynostosis, bone disease, or genetic syndromes. The authors also analyzed the presence of extracerebral fluid collection and ventricular asymmetry in children with UCS.

RESULTS

Fifty-six percent of patients had anterior right UCS and 44% had left-sided UCS. Type I UCS was seen in 1 patient, Type IIA in 20 patients, Type IIB in 20 patients, and Type III in 9 patients. The authors found that the nasion is usually deviated to the ipsilateral side of the synostosis, the bregma contralaterally, and the lambda ipsilaterally. The gap distances between the reference point and the SSS were 0–7.3 mm (mean 1.4 mm) at the nasion; 0–16.7 mm (mean 3.8 mm) at the midpoint nasion-bregma; 0–12 mm (mean 5.8 mm) at the bregma; 0–9.5 mm (mean 3 mm) at the midpoint bregma-lambda; and 0–11.6 mm (mean 5.5 mm) at the lambda. Conversely, a discrepancy of more than 1 mm between the SSS and the position of the suture was found only in 7 control cases (14%).

Of patients with UCS, 38% presented with an extracerebral fluid collection contralateral to the fused coronal suture. Fifty-two percent had a ventricular asymmetry, which was characterized by reduced ventricular volume ipsilateral to the synostosis in all but 1 patient.

CONCLUSIONS

In this study, the SSS was usually deviated contralaterally to the closed coronal suture. It tended to be in the midline of the cranial vault and could be projected virtually along an imaginary line passing through the midline of the cranial base. The authors recommend a distance of 37 mm from the sagittal suture as a safety margin during surgery.

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

✓The late failure of endoscopic third ventriculostomies (ETVs) has been described as a consequence of scarring phenomena involving the third ventricular floor. Although the possibility of closure of the stoma by intraventricular tumor seeding has been hypothesized, no case of ETV failure caused by tumor diffusion has been reported in the literature.

The authors report on a 19-year-old patient, affected by obstructive hydrocephalus associated with a periaqueductal pilocytic astrocytoma, who underwent late secondary lesional obstruction of an ETV performed at diagnosis. The authors suggest tumor extension along the diencephalic neural pathways as the possible cause of ETV failure in this patient.

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Alfredo Puca, Cesare Colosimo, Barbora Tirpakova, Libero Lauriola and Federico Di Rocco

✓ Extraaxial cavernous hemangiomas are rare intracranial lesions that can be located in different cranial compartments. Extension across different tissue planes such as the subcutaneous tissue, skull, orbital cavity, intracranial dura mater, and extracranial trigeminal divisions within the same patient has not been previously reported.

This 32-year-old woman suffered left exophthalmos, left sixth nerve palsy, and trigeminal neuropathy. Magnetic resonance imaging studies revealed an extensive multicompartmental lesion, with enhancement following Gd administration.

A left orbitopterional approach allowed removal of several cavernomatous lesions located in the orbit, frontotemporal dura, and lateral wall of the cavernous sinus. A histologically based diagnosis of extraaxial cavernous hemangioma was made. In the postoperative period the patient experienced a regression of her symptoms.

The authors report on a case of cavernous hemangioma with a unique extension to different intracranial/extracranial compartments. Although radical removal of the lesion was not feasible, partial excision allowed for satisfactory clinical control of the patient's symptoms.

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Federico Di Rocco, Carlos Eduardo Jucá, Eric Arnaud, Dominique Renier and Christian Sainte-Rose

Object

The aim of this study was to assess the efficacy of endoscopic third ventriculostomy (ETV) in the treatment of hydrocephalus in children with complex craniosynostosis.

Methods

Medical charts from 2194 children treated at the Craniofacial Unit of Necker Enfants Malades between 1995 and 2008 were reviewed. Among them, 293 were affected by a syndromic faciocraniosynostosis. The management of cases with hydrocephalus treated using ETV was examined and analyzed with regard to successes and failures.

Results

Forty-six children with associated progressive hydrocephalus were identified, and 11 (8 boys, 3 girls) had undergone an ETV. All were affected by a faciocraniosynostosis (Crouzon syndrome in 5, Pfeiffer syndrome in 3, and unclassifiable in 3). A descent of the cerebellar tonsils was found in 5 children. In 7 children the hydrocephalus was controlled by an ETV. In the remaining 4 cases a ventriculoperitoneal shunt was also needed. The delay between the 2 surgeries ranged from 1 week to 6 months in these 4 patients. The follow-up period ranged from 11 months to 13 years.

Conclusions

Performing an ETV may facilitate control of hydrocephalus associated with faciocraniosynostosis in select cases. However, because of the relatively high failure rate of this procedure, a close clinical and radiological monitoring of these children is mandatory.

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Marc Zanello, Thomas Baugnon, Alexandre Roux and Federico Di Rocco

OBJECTIVE

Breaking bad news is a difficult task in medical practice. Several breaking-bad-news training programs have been proposed. However, long-term results of such training have rarely been investigated. The aim of this study was to compare the short- and long-term evaluations by young neurosurgeons of a training program for breaking bad news to patients and their parents.

METHODS

Between 2012 and 2015, pediatric neurosurgery residents participated in a training day on breaking bad news in pediatric neurosurgery with professional actors. A personal debriefing, followed by a theoretical session, completed the training. Immediate feedback was evaluated through a survey administered at the end of the day. Long-term results were explored via an online form sent at least 3 years after the training completion.

RESULTS

Seventeen participants from 9 different countries were interviewed. Their immediate feedback confirmed their interest. For 71% of them, the program was very interesting, and 77% were extremely satisfied or very satisfied. All trainees wanted more training sessions. At a mean of 4.5 years of follow-up (range 3–6 years), 71% of the trainees fully remembered the session. Most of them (86%) reported a positive impact of the training on their career. Only 21% had another training session on breaking bad news during their residency. At long-term analysis, fewer trainees considered the duration of the training to have been sufficient (p = 0.044).

CONCLUSIONS

Breaking-bad-news training has a positive long-term educational impact even several years later. Such a training program should be implemented into pediatric neurosurgery residency.

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Federico Di Rocco, Matthew R. Garnett, Stephanie Puget, Francisco Pueyerredon, Thomas Roujeau, Francis Jaubert and Christian Sainte-Rose

✓Rosai–Dorfman disease (RDD) is a rare idiopathic histiocytic disorder that only occasionally involves the central nervous system (CNS). Previous cases of RDD involving the CNS were generally seen in adults. Pediatric cases of RDD are rare, and the disease in these cases typically has an indolent clinical course. In this report, the authors describe a pediatric case of intracranial RDD with rapid clinical and radiological progression.

A previously healthy 13-year-old girl presented with a 15-day history of progressive left-sided headaches, vomiting, and fever. On examination she was pyrexial but otherwise normal. Neuroimaging results demonstrated an extraaxial left frontal lesion with peripheral enhancement. A bur hole was drilled over the lesion to obtain a tissue sample and de-bulk the lesion. The initial histological results showed a nonspecific inflammatory lesion. Postoperatively, the patient was asymptomatic, and neuroimaging results confirmed a significant reduction in the size of the lesion. Repeated neuroimaging 3 months later, however, revealed a large recurrence of the lesion, which was removed macroscopically by a craniotomy. Histological analysis of the tissue confirmed the RDD diagnosis. At the latest follow-up (12 months) the patient had remained asymptomatic with no evidence of recurrence on neuroimaging. This is the first reported case of intracranial RDD with an aggressive clinical course.