Intracranial pressure patterns in children with craniosynostosis utilizing optical coherence tomography
Jordan W. SwansonWen XuGui-Shuang YingWei PanShih‐Shan LangGregory G. HeuerScott P. BartlettJesse A. Taylor
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Keywords:
Synostosis
Cranial vault
Remodelling the cranial vault in an attempt to increase the intracranial volume and thus control intracranial hypertension, whilst at the same time improving the patient's appearance, has been the mainstay of surgery for syndromic craniosynostosis. We report a case of craniosynostosis in whom cranial vault expansion was followed by the development of hind-brain herniation and hydrocephalus. This prompted a review of our other cases of craniosynostosis who had been evaluated by magnetic resonance imaging following surgery in order to assess the frequency of hind-brain herniation and hydrocephalus in these children. Magnetic resonance imaging had been performed in the postoperative evaluation of 34 cases of craniosynostosis who had undergone procedures intended to increase the intracranial volume. The position of the cerebellar tonsils and the presence or otherwise of hydrocephalus was recorded for all cases. The effectiveness of surgery in treating raised intracranial pressure (ICP) was evaluated by means of postoperative ICP monitoring and had been performed in 22 cases. Herniation of the hind-brain below the level of the foramen magnum was observed in 18 cases (53%). Hydrocephalus, requiring the insertion of a ventriculoperitoneal shunt, was present in 14 cases (41 %) and had developed after the cranial vault procedure in 9. The mean sleeping ICP measured postoperatively was normal (< 10 mm Hg) in 5, borderline (10–15) in 7, and raised (> 15 mm Hg) in 10 cases. Cranial vault expansion in complex craniosynostosis may fail to address the underlying aetiology of intracranial hypertension. Furthermore, both hydrocephalus and hind-brain herniation may develop following such surgery. Neither the increase in intracranial volume afforded by cranial vault expansion nor the shunting of hydrocephalus precludes the persistence of abnormal ICP. These findings are discussed in the light of possible mechanisms, in addition to cephalocranial disproportion responsible for intracranial hypertension in complex craniosynostosis. The implications for the surgical management of complex craniosynostosis are reviewed.
Cranial vault
Foramen magnum
Synostosis
Intracranial pressure monitoring
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After treating a child with familial sagittal craniosynostosis, clinocephaly, and bilateral parietomastoid/posterior squamosal suture fusion, the authors wondered if major-suture synostosis and clinocephaly were associated with abnormal fusion of minor lateral calvarial sutures.The authors reviewed all preoperative volume-rendered head computed tomography reconstructions performed for craniosynostosis at their institution from 2010 through 2014 and determined whether the sphenoparietal, squamosal, and parietomastoid sutures were open, partially fused, or fused. The authors determined whether any sutures were abnormally fused based upon a previous study from their center, in which abnormal fusion was defined as either 1 of 3 abnormal fusion patterns or abnormally-early fusion. The authors then determined the rate of abnormal fusion of these sutures and whether abnormal fusion was associated with (1) major-suture craniosynostosis, (2) type of craniosynostosis (sutures involved; single-suture versus multisuture; syndromic versus nonsyndromic), and (3) clinocephaly.In 97 included children, minor lateral sutures were abnormally fused in 8, or 8.2%, which was significantly higher than in children without craniosynostosis from our earlier study. Abnormal minor lateral suture fusion was not associated with the type of single-suture synostosis or with multisuture synostosis but was associated with syndromic synostosis. Four of 8 children with abnormal minor lateral suture fusion had multisuture synostosis and 6 had syndromic synostosis. Lateral sutures were abnormally fused in 1 of 4 subjects with clinocephaly, which was not significant.Abnormal minor lateral calvarial suture fusion is significantly associated with major-suture craniosynostosis, especially syndromic synostosis.
Synostosis
Sagittal suture
Dysostosis
Craniosynostoses
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Craniosynostoses
Dysostosis
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Coronal Suture Morphology and Synostotic Progression in Rabbits with Delayed-Onset Craniosynostosis.
Delayed-onset craniosynostosis has been reported in the human clinical literature and may represent part of the expanded phenotype of craniosynostosis. Delayed-onset synostosis (DOS) is also an integral part of the phenotypic spectrum in an inbred rabbit strain of craniosynostosis. The purpose of the present study was to qualitatively and quantitatively describe the perisynostotic events in the coronal sutures of rabbits with delay-onset coronal suture synostosis compared to age matched, wild-type control rabbits.
Coronal sutures from 80 rabbits (39 normal wild-type controls and 41 DOS) were collected for gross morphological and histological examination. Ages ranged from term (day 0) through 88 days of age. The sutures were harvested, formalin fixed, paraffin embedded, and stained with alizarin red for gross light microscopic analysis.
Qualitative results showed that by 25 days of age a dramatic thickening of the sutural ligament, an increase in the amount of bone in the osteogenic fronts, and an increase in the frequency of bony bridges between the frontal and parietal bones in rabbits with DOS compared to age matched, wild-type controls. No statistical significant differences in mean suture width or total suture area between the groups at the interdigitating zone were recorded at any age and cortical level (p> 0.05). At a region of the coronal suture closer to the midline, deemed the medial zone, a significant difference was found in mean suture width between groups at 88 day in age group at the ectocortical suture surface (p <.05); additionally, significant differences in mean suture area between the groups were recorded at age 25 and 88 (p <.05). A significant difference bony bridging frequency between the interdigitating zone and medial zone of DOS sutures was observed at the 45 day age group. The results of the 6X2 (age by group) two-way ANOVA indicate significant interaction at various cortical levels as well as total suture area at the interdigitating and medial zone (p < 0.05).
Results suggest that bony bridging and sutural ligament thickening occurs much earlier than coronal suture growth cessation in DOS rabbits and that these factors may limit subsequent sutural growth in an additive effect.
Coronal suture
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Frontal bone
Parietal bone
Sagittal suture
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The authors describe a comprehensive diagnostic algorithm,
individual pre-operative care and postoperative follow-up
procedures, established at the author’s workplace, to address
tailored pre-operative haematological preparation and cranial
vault remodelling surgery in craniosynostosis patients.
Materials and methodology: A set of 14 patients newly operated
upon using the remodelling technique is presented and compared
to a set of patients operated upon using strip craniectomy, in
terms of cosmetic effects, the need for transfusion, surgery
time and complications. Results: Remodellation technique
surgery patients showed significant improvement in cephalic
index and a better cosmetic effect compared with the strip
craniectomy patient group. There was no significant difference
in surgery time between the operational techniques.
Pre-operative haematological preparation was sufficient to
eliminate the higher transfusion requirements of very young
patients. Conclusions: The remodelling surgery technique was
found to provide better cosmetic and therapeutic effects
compared with strip craniectomy. Cranial vault remodelling
surgery combined with comprehensive, tailored pre-operative
care is a safe and efficient procedure in craniosynostosis
treatment even in very young children.
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This chapter, provides an excellent review of a core pediatric surgical topic; craniosynostosis and cranial vault remodeling. The authors describe the pathophysiology of the four main cranial sutures and the characteristics associated with each of their premature closures. The syndromes associated with the synostoses are reviewed. The perioperative considerations for cranial vault reconstruction are presented.
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Craniosynostoses
Vault (architecture)
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Synostosis
Dysostosis
Craniosynostoses
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Craniosynostosis is a congenital disease which consists of premature fusion of one or more cranial sutures, resulting in an abnormal head shape. Patients are usually treated by cranial vault expansion surgery to minimize the potential for brain damage. Full thickness cranial defects result from the expansion surgery, with the size directly proportional to the degree of expansion. The growing cranial skeleton has a unique regenerative capacity to heal small defects; however, when this regenerative capacity is exceeded, the defect is classed as one of critical size and requires surgical treatment to restore protection to the underlying brain. Although what constitutes a critical cranial defect is well known in animal models, it is not as clear for pediatric human skulls. The purpose of this study is to investigate a method that can effectively quantify healing of the pediatric cranial defect surface after cranial vault expansion surgery for craniosynostosis.
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Abstract A considerable amount of information is available on various types of craniosynostoses. The patient exhibiting single suture synostosis that progresses to involve multiple sutures is distinctly uncommon, as is the patient exhibiting delayed synostosis involving all of the calvarial sutures. We report a group of 11 such patients with progressive and delayed holocalvarial synostosis. Most patients exhibited features of raised intracranial pressure or developmental delay, and in all patients symptoms were relieved after surgery. The diagnostic and therapeutic implications of this type of presentation in craniosynostosis are discussed.
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Craniosynostoses
Presentation (obstetrics)
Dysostosis
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Craniosynostosis describes a fusion of one or more sutures in the skull. It can occur in isolation or as part of a syndrome. In either setting, it is a condition which may lead to raised intracranial pressure. The exact cause of raised intracranial pressure in craniosynostosis is unknown. It may be due to; a volume mismatch between the intracranial contents and their containing cavity, venous hypertension, hydrocephalus or airway obstruction, which is often a sequela of an associated syndrome. At Great Ormond Street Hospital, after hydrocephalus and airway obstruction have been treated, the next surgical treatment of choice is cranial vault expansion. This expansion has been shown to reduce intracranial pressure, interestingly despite its success, the reasons behind its benefits are not fully understood. Using reconstructed 3-dimensional imaging, accurate measurement of cranial volumes can now be achieved. The aim of this project is to use the advances in 3-dimensional imaging and image processing to provide novel information on the volume changes that occur following cranial vault expansion. This information will be combined with clinical metrics to create a greater understanding of the causes of raised intracranial pressure in craniosynostosis, why cranial vault expansion treats them and whether there is an optimal volume expansion.
Cranial vault
Crouzon syndrome
Intracranial pressure monitoring
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