Venous-plexus-associated lymphoid hubs support meningeal humoral immunity.

There is increasing interest in how immune cells in the meninges-the membranes that surround the brain and spinal cord-contribute to homeostasis and disease in the central nervous system1,2. The outer layer of the meninges, the dura mater, has recently been described to contain both innate and adaptive immune cells, and functions as a site for B cell development3-6. Here we identify organized lymphoid structures that protect fenestrated vasculature in the dura mater. The most elaborate of these dural-associated lymphoid tissues (DALT) surrounded the rostral-rhinal confluence of the sinuses and included lymphatic vessels. We termed this structure, which interfaces with the skull bone marrow and a comparable venous plexus at the skull base, the rostral-rhinal venolymphatic hub. Immune aggregates were present in DALT during homeostasis and expanded with age or after challenge with systemic or nasal antigens. DALT contain germinal centre B cells and support the generation of somatically mutated, antibody-producing cells in response to a nasal pathogen challenge. Inhibition of lymphocyte entry into the rostral-rhinal hub at the time of nasal viral challenge abrogated the generation of germinal centre B cells and class-switched plasma cells, as did perturbation of B-T cell interactions. These data demonstrate a lymphoid structure around vasculature in the dura mater that can sample antigens and rapidly support humoral immune responses after local pathogen challenge.

Resilient anatomy and local plasticity of naive and stress haematopoiesis.

The bone marrow adjusts blood cell production to meet physiological demands in response to insults. The spatial organization of normal and stress responses are unknown owing to the lack of methods to visualize most steps of blood production. Here we develop strategies to image multipotent haematopoiesis, erythropoiesis and lymphopoiesis in mice. We combine these with imaging of myelopoiesis1 to define the anatomy of normal and stress haematopoiesis. In the steady state, across the skeleton, single stem cells and multipotent progenitors distribute through the marrow enriched near megakaryocytes. Lineage-committed progenitors are recruited to blood vessels, where they contribute to lineage-specific microanatomical structures composed of progenitors and immature cells, which function as the production sites for each major blood lineage. This overall anatomy is resilient to insults, as it was maintained after haemorrhage, systemic bacterial infection and granulocyte colony-stimulating factor (G-CSF) treatment, and during ageing. Production sites enable haematopoietic plasticity as they differentially and selectively modulate their numbers and output in response to insults. We found that stress responses are variable across the skeleton: the tibia and the sternum respond in opposite ways to G-CSF, and the skull does not increase erythropoiesis after haemorrhage. Our studies enable in situ analyses of haematopoiesis, define the anatomy of normal and stress responses, identify discrete microanatomical production sites that confer plasticity to haematopoiesis, and uncover unprecedented heterogeneity of stress responses across the skeleton.

Multimodality imaging reveals angiogenic evolution in vivo during calvarial bone defect healing.

The healing of calvarial bone defects is a pressing clinical problem that involves the dynamic interplay between angiogenesis and osteogenesis within the osteogenic niche. Although structural and functional vascular remodeling (i.e., angiogenic evolution) in the osteogenic niche is a crucial modulator of oxygenation, inflammatory and bone precursor cells, most clinical and pre-clinical investigations have been limited to characterizing structural changes in the vasculature and bone. Therefore, we developed a new multimodality imaging approach that for the first time enabled the longitudinal (i.e., over four weeks) and dynamic characterization of multiple in vivo functional parameters in the remodeled vasculature and its effects on de novo osteogenesis, in a preclinical calvarial defect model. We employed multi-wavelength intrinsic optical signal (IOS) imaging to assess microvascular remodeling, intravascular oxygenation (SO2), and osteogenesis; laser speckle contrast (LSC) imaging to assess concomitant changes in blood flow and vascular maturity; and micro-computed tomography (µCT) to validate volumetric changes in calvarial bone. We found that angiogenic evolution was tightly coupled with calvarial bone regeneration and corresponded to distinct phases of bone healing, such as injury, hematoma formation, revascularization, and remodeling. The first three phases occurred during the initial two weeks of bone healing and were characterized by significant in vivo changes in vascular morphology, blood flow, oxygenation, and maturity. Overall, angiogenic evolution preceded osteogenesis, which only plateaued toward the end of bone healing (i.e., four weeks). Collectively, these data indicate the crucial role of angiogenic evolution in osteogenesis. We believe that such multimodality imaging approaches have the potential to inform the design of more efficacious tissue-engineering calvarial defect treatments.

[Topographic study of the parietal region for the elaboration of revascularized cranium vault autograft]. / Izuchenie topografo-anatomicheskikh osobennostei temennoi oblasti pri razrabotke revaskulyariziruemogo kostno-fastsial'nogo temennogo autotransplantata.

OBJECTIVE: Topographic and blood vessel architecture study of the parietal area and distal regional pool of the superficial temporal artery (STA) to assess the possibility of revascularized cranium vault bone autograft formation. MATERIAL AND METHODS: For the topographic and anatomical study, 30 non-fixed corpses (17 male and 13 female) were selected, the average age of which was 59±5 years. In the anamnesis and catamnesis, there were no indications of trauma or other pathology of the head and neck, including vascular. STA was contrasted with a non-radiocontrast dye (brilliant green) with the introduction of the dye into the STA with preliminary ligation of the frontal branch of the STA. The area of blood supply to soft tissue and bone structures was studied. The angioarchitectonics of the parietal region was studied, the feeding vessel of the studied flap was identified. RESULTS: The obtained anatomical landmarks for the collection of CPFP flap make it possible to form a flap with high accuracy and minimize the morbidity of the donor area.

Cranial diploic channels and their veins - a review of literature.

The current paper is a review of the results attained in the past and current anatomical studies, aimed at understanding the variability and function of the diploic venous system of the human skull. The diploic veins can serve as transit for infections from the scalp to the structures contained within the cranial cavity via the emissary veins, due to their interconnections with the pericranial veins, meningeal veins and dural sinuses. Thereby this clinical aspect has also been discussed.

Like a hole in the head: Development, evolutionary implications and diseases of the cranial foramina.

Cranial foramina are holes in the skull through which nerves and blood vessels pass to reach both deep and superficial tissues. They are often overlooked in the literature; however they are complex structures that form within the developing cranial bones during embryogenesis and then remain open throughout life, despite the bone surrounding them undergoing constant remodelling. They are invaluable in assigning phylogeny in the fossil record and their size has been used, by some, to imply function of the nerve and/or blood vessel that they contained. Despite this, there are very few studies investigating the development or normal function of the cranial foramina. In this review, we will discuss the development of the cranial foramina and their subsequent maintenance, highlighting key gaps in the knowledge. We consider whether functional interpretations can be made from fossil material given a lack of knowledge regarding their contents and maintenance. Finally, we examine the significant role of malformation of foramina in congenital diseases such as craniosynostosis.

Effect of silicon-doped calcium phosphate cement on angiogenesis based on controlled macrophage polarization.

Vascularization is an important early indicator of osteogenesis involving biomaterials. Bone repair and new bone formation are associated with extensive neovascularization. Silicon-based biomaterials have attracted widespread attention due to their rapid vascularization. Although calcium phosphate cement (CPC) is a mature substitute for bone, the application of CPC is limited by its slow degradation and insufficient promotion of neovascularization. Calcium silicate (CS) has been shown to stimulate vascular endothelial proliferation. Thus, CS may be added to CPC (CPC-CS) to improve the biocompatibility and neovascularization of CPC. In the early phase of bone repair (the inflammatory phase), macrophages accumulate around the biomaterial and exert both anti- and pro-inflammatory effects. However, the effect of CPC-CS on macrophage polarization is not known, and it is not clear whether the effect on neovascularization is mediated through macrophage polarization. In the present study, we explored whether silicon-mediated macrophage polarization contributes to vascularization by evaluating the CPC-CS-mediated changes in the immuno-environment under different silicate ion contents both in vivo and in vitro. We found that the silicon released from CPC-CS can promote macrophage polarization into the M2 phenotype and rapid endothelial neovascularization during bone repair. Dramatic neovascularization and osteogenesis were observed in mouse calvarial bone defects implanted with CPC-CS containing 60% CS. These findings suggest that CPC-CS is a novel biomaterial that can modulate immune response, promote endothelial proliferation, and facilitate neovascularization and osteogenesis. Thus, CPC-CS shows potential as a bone substitute material.

Stromal cell-derived factor (SDF)-1α and platelet-rich plasma enhance bone regeneration and angiogenesis simultaneously in situ in rabbit calvaria.

The current study aimed to evaluate the effects of chemokine stromal cell-derived factor (SDF)-1α and platelet-rich plasma (PRP) on bone formation and angiogenesis, and to assess whether SDF-1α and PRP could function synergistically. Four evenly distributed defects (8 mm in diameter) were generated in the calvarial bones of New Zealand white rabbits. All rabbits received four treatment regimens containing autogenous bone particles (AB), SDF-1α, or PRP. AB group presented significantly less bone formation compared with the other three groups 2 and 4 weeks after surgery. The amount of newly formed bone in the AB+PRP+SDF-1α group was similar to that in the AB + SDF-1α group at the 4-week time-point (p = 0.65), and was much greater than that in the AB and AB+PRP group (p < 0.001). Meanwhile, more new blood vessels were formed in the AB+PRP, AB+SDF-1α, and AB+PRP+SDF-1α group versus the AB group. AB+PRP+SDF-1α group showed statistically increased angiogenesis compared with the AB+PRP and AB+SDF-1α groups (both p < 0.05) after treatment for 2 and 4 weeks. These findings indicated that SDF-1α and PRP might exhibit synergistic effects to promote angiogenesis in early bone regeneration.

Calvarial diploic venous channels: delineation with maximal intensity projection technique.

PURPOSE: To date, very few studies have explored the three-dimensional architecture of calvarial diploic venous channels (CDVCs). This study aimed to characterize the three-dimensional architecture of CDVCs using maximum intensity projection (MIP) images based on contrast-enhanced magnetic resonance imaging (MRI). METHODS: A total of 77 patients with intact calvarial hemispheres and underlying dura mater and dural sinuses underwent contrast-enhanced MRI. Among them, we extracted the data of 49 with at least a part of the major CDVC pathways identified on the MIP images for analysis. RESULTS: On serial contrast-enhanced MRI images, the CDVCs were commonly detected as curvilinear structures with inhomogeneous diameters and tributaries, while the MIP images delineated the three-dimensional architecture of the developed CDVC pathways. More than such CDVC pathway was entirely delineated on the right in 67.3% and on the left in 71.4%, most frequently in the frontal and temporal regions, with their connecting sites to the sphenoparietal and superior sagittal sinuses. The morphology, distribution, and course of the identified CDVCs were highly variable. In 55.1%, the CDVCs formed fenestrations that were variable in size, shape, and number. CONCLUSIONS: The developed CDVC pathways may be characterized by morphological variability and fenestrations. Thin-sliced, contrast-enhanced MRI is useful to depict diploic veins, while MIP images allow for better appreciation of the entire course of the developed CDVC pathways. Traumatic and intraoperative disconnection between the dura mater overlying the dural sinuses and the adjacent inner table of the skull can cause epidural venous bleeding.

Ontogenetic changes of diploic channels in modern humans.

OBJECTIVES: The diploic channels are bony passages of veins, running within frontal, parietal, and occipital bones. In this study, we investigate ontogenetic changes of these channels in a sample of nonadult and adult modern humans. MATERIALS AND METHODS: Using computed tomography scans of dried crania, we provide quantitative comparisons of lumen size, branch length, volume, and vascular asymmetries, and correlations with age, cranial size, and bone thickness. RESULTS: The vascular system displays progressive but nonlinear changes throughout ontogeny, becoming even more complex with adulthood. Vascular variables are significantly different in frontal, parietal, and occipital bones for most of the postnatal ontogeny. Diploic channels of the left and right sides are developed similarly. Vascular variables display a nonlinear association with age and cranial size in modern humans. Cranial bone thickness is shown to be a major determinant of lumen size, branch length, and volume. CONCLUSIONS: A previous radiographic survey suggested that diploic channels are more developed in adult modern humans than in nonadults. Recent advances in digital anatomy have been used in this study to investigate this craniovascular structure. The complexity of the channels increases during development, with a noticeable boost in adults. Taking into account the potential metabolic differences and constraints associated with modern human brain size and shape, the vascular differences found might be related to endocranial thermoregulation.

Surgical Decision-Making in Microvascular Reconstruction of Composite Scalp and Skull Defects.

OBJECTIVES: Microvascular reconstruction of composite scalp and skull defects requires careful planning of both cranial bone and soft-tissue coverage. The current body of literature has yet to identify a "best practice" approach to achieve these goals. METHODS: A retrospective chart review was performed. Patients with composite defects who underwent combined microvascular surgery of the scalp and skull were included over a 6-year period. Reconstructions were classified by: microvascular flap, cranioplasty, timing of cranioplasty (primary or delayed), and exposure to radiation. RESULTS: Forty-five microvascular flaps were performed for 36 patients. Fasciocutaneous flaps were more likely to experience complications than other microvascular flaps (50.0% versus 8.6%, P = 0.008). Forty of the 50 patients (80%) underwent cranioplasty, including 19 autogenous and 21 alloplastic reconstructions. There were 8 total complications: partial flap loss with implant exposure (n = 5), cranioplasty infection (n = 2), and wound dehiscence (n = 1). Alloplastic implants experienced more frequent complications than autologous reconstructions (33.3% versus 5.3%, P = 0.046). Titanium implants demonstrated the higher rates of complications than other groups (P = 0.014). Titanium implants also had more complications relative to poly-ether-ether-ketone implants (60.0% versus 9.1%, P = 0.024). Immediate alloplastic cranioplasty was associated with a significant increase in complications relative to autogenous reconstruction (54.5% versus 5.5%, P = 0.027), and no significant difference in the delayed group (10% versus 0%, P = 0.740). CONCLUSION: According to authors' knowledge, myofascial flaps yield the lowest complication rate and when possible, autologous cranioplasty is preferred. When defects are too large to accommodate autogenous bone, the authors prefer delayed prefabricated poly-ether-ether-ketone implant reconstruction.

Elective Sonolucent Cranioplasty for Real-Time Ultrasound Monitoring of Flow and Patency of an Extra- to Intracranial Bypass.

Cross-sectional imaging studies or catheter angiogram are the imaging modalities of choice to evaluate bypass patency after extra- to intracranial (EC-IC) bypass surgery. Although providing accurate results, these imaging modalities are time-consuming and/or present radiation risk for the patient. Ultrasound imaging is a fast and widely available imaging modality, but is limited in this setting due to the non-sonolucent autologous bone flap covering the bypass after surgery. The recently FDA approved clear polymethyl methacrylate (PMMA) cranioplasty implant overcomes this limitation by its sonolucent characteristic, but has not yet been used in the setting of EC-IC bypass surgery. Here, the authors describe for the first time the feasibility of an elective sonolucent cranioplasty to monitor flow and patency of an EC-IC bypass in real time using ultrasound. This moyamoya patient underwent a direct superficial temporal artery to middle cerebral artery (STA-MCA) bypass, after which a PMMA implant was used to close the craniotomy defect, instead of reimplanting the autologous bone flap. Immediate postoperative bedside transcranioplasty ultrasound confirmed bypass patency and allowed for quantitative flow measurements as well as for exclusion of postoperative hemorrhage. Postoperative CTA and catheter angiogram confirmed patency of the bypass without complications. This report shows for the first time that this technique is feasible and permits bedside transcranioplasty ultrasound assessment of bypass flow in real time, confirmed with angiography. This technique may permit easy comparison of baseline findings with follow up assessments and facilitate less invasive monitoring of bypass patency.

Cranial burr hole with erythropoietin administration induces reverse arteriogenesis from the enriched extracranium.

It is challenging to revitalize ischemic penumbra after an acute stroke with intracranial perfusion insufficiency. To evaluate whether cranial burr hole and erythropoietin (EPO) generate effective revascularization, we investigated the efficacy of the augmentation method for reverse arteriogenesis from the healthy extracranial milieu. An intracranial perfusion insufficiency was created through bilateral internal carotid artery ligation (bICAL) in Sprague-Dawley rats. We administered recombinant human EPO (5000 U/kg) or saline intraperitoneally for 3 days after bICAL. Mechanical barrier disruption (MBD) was performed through a cranial burr hole with small dural cracks in the right hemisphere. The ipsilateral hemisphere with MBD grossly showed vascular networks between the extra- and intra-cranial spaces 2 weeks after the MBD procedure. It also showed significantly increased vessels in the intracranial vasculature adjacent to the MBD region (p = 0.0006). The levels of pro-angiogenic and inflammatory factors with prominent markers of vessel permeability were also significantly increased (MBD-only vs. control; Tnf-α, p = 0.0007; Vegf, p = 0.0206). In the EPO-administered group, such elevations in inflammation were significantly mitigated (combined vs. MBD-only; Tnf-α, p = 0.0008). The ipsilateral hemisphere with MBD-EPO (vs. MBD-only) showed significantly increased vessels (RECA-1, p = 0.0182) and their maturation (RECA-1/α-SMA, p = 0.0046), with upregulation of tumor growth factor-ß1 (Tgf-ß1, p = 0.037) and matrix metalloproteinase-2 (Mmp-2, p = 0.0488). These findings were completely blocked by minocycline (MIC) administration during in vivo (Tgf-ß1, p = 0.0009; Mmp-2, p < 0.0001) and in vitro experiments (tube formation, p < 0.0001). Our data suggest that the MBD procedure (for angiogenic routes) and EPO administration (for an arteriogenic booster) are complimentary and can facilitate successfully "reverse arteriogenesis" in subjects with intracranial perfusion insufficiency.

Application of optical flow algorithms to laser speckle imaging.

Since of its introduction in 1980s, laser speckle imaging has become a powerful tool in flow imaging. Its high performance and low cost made it one of the preferable imaging methods. Initially, speckle contrast measurements were the main algorithm for analyzing laser speckle images in biological flows. Speckle contrast measurements, also referred as Laser Speckle Contrast Imaging (LSCI), use statistical properties of speckle patterns to create mapped image of the blood vessels. In this communication, a new method named Laser Speckle Optical Flow Imaging (LSOFI) is introduced. This method uses the optical flow algorithms to calculate the apparent motion of laser speckle patterns. The differences in the apparent motion of speckle patterns are used to identify the blood vessels from surrounding tissue. LSOFI has better spatial and temporal resolution compared to LSCI. This higher spatial resolution enables LSOFI to be used for autonomous blood vessels detection. Furthermore, Graphics Processing Unit (GPU) based LSOFI can be used for quasi real time imaging.

Clinical and Molecular Study of ELMO-2-Related Massive Intraosseous Vascular Malformations: Lessons Learned From 25 Years of Follow-up.

Massive intraosseous vascular malformations, a relatively rare entity in the vascular malformation spectrum, deserves attention as involving the membranous bones of the craniofacial skeleton and may lead to severe life-threatening hemorrhages and even death. The main aim of this study was to summarize the 25 years of clinical experience with these vascular malformation osseous patients, focus on the molecular and genetic aspect of the clinical entity, and to emphasize the certain challenging conditions in the treatment of these patients. All the patients appeared to be unaffected at birth, whereas initial symptoms occasionally began with painless swelling in the mandible in early childhood. The disease was progressive in behavior especially in the pubertal ages and was specifically involving the maxilla and mandible of the craniofacial skeleton in all the patients. Calvarium and cranial base were the second most common involved regions among these patients (62.5%). Clavicular (50%), costal (25%), and vertebral (25%) involvements were also a significant manifestation of the disease. Tissue samples obtained from the affected individuals and the blood samples from their families were matched, revealing a loss of function mutation in the ELMO-2 gene of chromosome 20 leading to developmental abnormality of the vascular structures via RAC1 signaling and leading to abnormally enlarged vessels in the intraosseus portion of the membranous bone. Immunohistochemical staining revealed positive CD31 and smooth muscle actin staining but negative proliferation and maturity markers such as Ki-67, desmin, h-caldesmon, and myofibroblast-like desmin. The follow-up of 3 of 5 patients ended up with mortality (60%). vascular malformation osseous is intraosseous vascular malformation with aggressive biological behavior associated with ELMO-2 gene mutation. Further studies for obtaining prenatal diagnosis and achievement of gene therapy should take place. As the disease rapidly progresses as the affected individual grows, surgical interventions should be taken into consideration before the initiation of complications.

Reconstruction of composite defects of the scalp and neurocranium-a treatment algorithm from local flaps to combined AV loop free flap reconstruction.

BACKGROUND: Reconstruction of cranial composite defects, including all layers of the scalp and the neurocranium, poses an interdisciplinary challenge. Especially after multiple previous operations and/or radiation therapy, sufficient reconstruction is often only possible using microsurgical free flap transplantation. The aim of this study was to analyze the therapy of interdisciplinary cases with composite defects including the scalp and neurocranium. METHODS: From 2009 to 2017, 23 patients with 18 free flaps and 10 pedicled/local flaps were analyzed. First choices for free flaps were muscle flaps followed by fasciocutaneous flaps. RESULTS: Except for four patients, a stable coverage could be reached in the first operation. Three of these patients received a local scalp rotation flap in the first operation and needed an additional free flap because the local flap was no longer sufficient for coverage after wound healing deficiency or tumor relapse. The superficial temporal artery or external carotid artery served as recipient vessels. In special cases, venous grafts or an arteriovenous loop (AV loop) were used as extensions for the recipient vessels. CONCLUSIONS: In summary, an interdisciplinary approach with radical debridement of infected or necrotic tissue and the reconstruction of the dura mater are essential to reach a stable, long-lasting reconstructive result. Based on our experience, free flaps seem to be the first choice for patients after multiple previous operations and/or radiation therapy.

Sickle cell crisis: A crisis of a different sort?

A 9-year-old boy with sickle cell disease (SCD) was presented to the emergency department with acute headache and swelling over his bilateral temporoparietal region. There was no history of antecedent trauma, fever, vomiting or other features of an intercurrent illness. On arrival, his blood pressure was 112/62 mm Hg, heart rate was 98/min and his Glasgow Coma Scale score was 15/15. There was evidence of significant scalp tenderness over the bilateral temporoparietal region. A complete neurological examination including direct and consensual pupillary response was unremarkable. Initial investigations revealed haemoglobin of 9.6 g/dL, leucocyte count of 6.8/mm3, platelet count of 219/mm3 and a normal coagulation profile. His current medications included hydroxyurea and penicillin prophylaxis. He underwent an urgent CT of the head followed by MRI of the brain, which revealed abnormalities as depicted in figures 1,2 edpract;103/6/290/F1F1F1Figure 1CT head. edpract;103/6/290/F2F2F2Figure 2MRI brain. QUESTION 1: Is this one of the most common neurological presentation seen in sickle cell crisis? QUESTION 2: How common is this presentation in paediatric SCD? QUESTION 3: What is the best way to manage this child?

Cortical bridging venous segment protruding into the inner skull depression: a potential site of hemorrhage at external ventriculostomy.

PURPOSE: The cortical bridging venous segment protruding into the inner skull depression (CBVISD), a distinctive structure found in the calvarial convexity, may be a potential risk factor for hemorrhagic complications resulting from trephination. However, this subject has not been documented. In the present study, we explore the CBVISD using magnetic resonance imaging (MRI). METHODS: A total of 94 patients underwent contrast-enhanced MRI. Distributions of the CBVISDs were recorded in two symmetrical regions of the frontal calvarial convexity. The lateral dimensions on both hemispheres were defined to range between 25 and 35 mm from the midsagittal section. The anteroposterior axis was delimited anteriorly by the supraorbital bar and posteriorly by the coronal suture, which was further divided into four parts: the forehead (FH), anterior frontal (AF), FH-AF junctional, and posterior frontal (PF) regions. RESULTS: Analysis of the delineations revealed that the CBVISDs had variable shapes and sizes. They were identified on the right side in 40% of the 94 patients and in 41% on the left side. Among the 91 identified CBVISDs, 5.5% of the CBVISDs were located in the FH, 17.6% in the FH-AF, 34.1% in the AF, and 42.9% in the PF. CONCLUSIONS: The CBVISDs are most frequently located at the common trephination site of external ventriculostomy. Trephination performed in the FH and FH-AF junctional regions may be safer than that in more posterior frontal areas.

[Three-Dimensional Skull Model with Vascular Structures for Occipital Expansion in Patients with Developed Occipital Emissary Veins].

We report the application of a three-dimensional skull model with vascular structures for occipital expansion in a patient who had developed occipital emissary veins. A 15-year-old boy visited a dentist for orthodontic treatment and was diagnosed with midface hypoplasia. He had undergone a ventriculo-peritoneal shunt at the age of 10 months. On visiting our institution, he was diagnosed with exophthalmos, low visual acuity, reversed bite, and developmental retardation. Moreover, he had sleep apnea and was supported with continuous positive airway pressure at night. Computed tomography and magnetic resonance imaging demonstrated brachycephalus, chronic tonsillar herniation, midface hypoplasia, and well-developed occipital emissary veins. We chose occipital expansion for lowering intracranial pressure and created a three-dimensional skull model with vascular structures and shunt tube. On this model, we wrote scalp incision and craniotomy lines avoiding injury to the veins and shunt tube. The operation was completed using this plan. Patients with syndromic craniosynostosis frequently have developed emissary veins and previous shunt operation. The three-dimensional skull model with vascular structures will be expected to reduce the risks of surgery.