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Alexander E. Ropper, Gary F. Rogers, Emily B. Ridgway and Mark R. Proctor

The authors report the case of a large idiopathic frontal bone defect and concomitant sagittal synostosis corrected by autologous exchange cranioplasty by using a corticocancellous bone graft and cranial vault expansion.

An otherwise healthy, developmentally normal 6-year-old girl presented to our clinic with a large frontal bone defect. The osseous defect was midline and inferior to the coronal sutures, and the underlying dura was slightly tense. She had no signs or symptoms of increased intracranial pressure, and her head circumference and cephalic index were normal. Imaging demonstrated fusion of the sagittal synostosis.

The defect was repaired using full-thickness autologous bone harvested from the bilateral parietal regions, which were widened using barrel-stave osteotomies to reduce pressure on the graft site in the setting of sagittal synostosis and mild cranial narrowing. The donor sites were covered with autologous particulate bone graft harvested from the endocortical surface of the grafted segments and the ectocortical surface of the intact parietal bones. The donor and recipient sites healed. Imaging revealed that the particulate bone healed with a thickness similar to the surrounding bone.

This bony defect is analogous to parietal foramina and may have a similar etiopathogenesis. The technique of autologous exchange cranioplasty using corticocancellous particulate bone graft provides a simple and reliable method to repair large structural calvarial defects.

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John Berry-Candelario, Emily B. Ridgway, Ronald T. Grondin, Gary F. Rogers and Mark R. Proctor

Object

The primary goals of treatment in the infant with craniosynostosis are to correct the deformity and allow for adequate brain growth in as safe and effective a manner as possible. Herein, the authors present the results of treating craniosynostosis using an endoscope-assisted strip craniectomy and postoperative helmet therapy (EASC + PHT) in the hopes of providing further evidence of its role in the treatment of multiple different forms of craniosynostosis. This is a retrospective review of the patients treated with this technique at Children's Hospital Boston.

Methods

The electronic medical records of all children with craniosynostosis treated using this technique were reviewed retrospectively. A priori, data were collected for deformity type, patient age at surgery, number of transfusions, operative time, length of hospital stay, and anthropometric measurements.

Results

One hundred seventy-three patients (61 females and 112 males) were treated at our institution between July 2004 and March 2011 with EASC + PHT. The mean operative time was 46.30 minutes. Eight (4.6%) of the 173 patients received blood transfusions. The average length of hospital stay was 1.35 days, with the majority of patients being discharged the day after surgery. All complications and any patient who required additional craniofacial reconstructions are discussed. In addition, a subgroup analysis was done for patients who had undergone surgery and had longer than 1 year of follow-up.

Conclusions

The authors' growing database of patients supports the experiences described by others that early treatment of craniosynostosis with an EASC + PHT is a safe and efficacious technique. In addition, cost reduction due to decreased hospital stay and limitation of blood transfusions are demonstrable benefits associated with the use of this technique.

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Benjamin C. Wood, Albert K. Oh, Robert F. Keating, Michael J. Boyajian, John S. Myseros, Suresh N. Magge and Gary F. Rogers

OBJECT

Progressive postnatal pansynostosis (PPP) is a rare form of craniosynostosis that is characterized by a normal head shape, insidious decrease in percentile head circumference, and high rates of elevated intracranial pressure (ICP). This investigation describes the clinical, radiographic, and genetic features of this entity.

METHODS

The authors’ craniofacial database for the period 1997–2013 was retrospectively culled to identify patients who had a normal or near-normal head shape and CT-confirmed multiple-suture synostosis. Patients with kleeblatt-schädel or previous craniofacial surgery were excluded. All demographic information was collected and analyzed.

RESULTS

Seventeen patients fit the inclusion criteria. Nine patients had a syndromic diagnosis: Crouzon syndrome (n = 4), Pfeiffer syndrome (n = 2), Saethre-Chotzen syndrome (n = 1), Apert syndrome (n = 1), and achondroplasia (n = 1). With the exception of 3 patients with mild turricephaly, all patients had a relatively normal head shape. Patients were diagnosed at an average age of 62.9 months. Nearly all patients had some combination of clinical, radiographic, or ophthalmological evidence of increased ICP.

CONCLUSIONS

PPP is insidious; diagnosis is typically delayed because the clinical signs are subtle and appear gradually. All normocephalic infants or children with a known or suspected craniosynostotic disorder should be carefully monitored; any decrease in percentile head circumference or signs/symptoms of increased ICP should prompt CT evaluation.

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Emily B. Ridgway, John Berry-Candelario, Ronald T. Grondin, Gary F. Rogers and Mark R. Proctor

Object

Suturectomy as a treatment for craniosynostosis was largely replaced in the late twentieth century by more extensive, but predictable, cranial remodeling procedures. Recent technical innovations, such as using the endoscope combined with postoperative orthotic reshaping, have led to a resurgence of interest in suturectomy as a safer, less invasive method.

Methods

A retrospective chart review was performed for all cases of sagittal synostosis treated with endoscopic sagittal suture strip craniectomy and helmet therapy between 2004 and 2008. Data collected included gestational age, genetic evaluations and syndromic status, age at operation, duration of procedure, need for blood transfusions, length of hospital stay, preoperative and postoperative head circumference percentile and cranial index, duration of helmet use, length of follow-up, complications, and revisions.

Results

Fifty-six patients with isolated sagittal synostosis were treated using endoscopic suturectomy and completed helmet therapy. Mean age at time of procedure was 3.24 months. Mean operative duration was 45.32 minutes. Mean hospital stay was 1.39 days. There were 2 transfusions and no deaths. The mean length of follow-up was 2.34 years. Helmet therapy was instituted for a mean of 7.47 months. Head circumference percentile increased from 61.42% to 89.27% over 2 years of follow-up. Cranial index increased from a preoperative mean of 0.69 to 0.76 over 2 years of follow-up. Reoperations for synostosis included 1 sagittal suture refusion and 2 cases in which other sutures fused.

Conclusions

Sagittal synostosis can be safely treated with endoscopic suturectomy and helmet therapy. Improvements in cranial volume and shape are comparable to open procedures and are enduring.

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Manish K. Kasliwal, Gary F. Rogers, Shakti Ramkissoon, Alexander Moses-Gardner, Kyle C. Kurek and Edward R. Smith

Psammomatoid ossifying fibroma (POF), a variant of ossifying fibroma, is a benign fibroosseous lesion typically arising within the nasal cavity, paranasal sinuses, and orbit. Cranial vault involvement is exceedingly rare, with very few cases reported in the literature. The authors report a case of POF in the neurocranium of an 11-year-old child, 4 years after chemotherapy and radiation therapy for acute lymphoblastic leukemia. This case is reported in view of its rarity, novelty of presentation, and the difficulty in diagnosis due to its radiological resemblance to aneurysmal bone cyst or monostotic cystic fibrous dysplasia, further aggravated by the clinical scenario. A novel technique of cranial reconstruction called autologous particulate exchange cranioplasty was used following tumor excision.

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Benjamin C. Wood, Edward S. Ahn, Joanna Y. Wang, Albert K. Oh, Robert F. Keating, Gary F. Rogers and Suresh N. Magge

OBJECTIVE

Endoscopic strip craniectomy (ESC) with postoperative helmet orthosis is a well-established treatment option for sagittal craniosynostosis. There are many technical variations to the surgery ranging from simple strip craniectomy to methods that employ multiple cranial osteotomies. The purpose of this study was to determine whether the addition of lateral barrel-stave osteotomies during ESC improved morphological outcomes.

METHODS

An IRB-approved retrospective review was conducted on a consecutive series of cases involving ESC for sagittal craniosynostosis at 2 different institutions from March 2008 to August 2014. The patients in Group A underwent ESC and those in Group B had ESC with lateral barrel-stave osteotomies. Demographic and perioperative data were recorded; postoperative morphological outcomes were analyzed using 3D laser scan data acquired from a single orthotic manufacturer who managed patients from both institutions.

RESULTS

A total of 73 patients were included (34 in Group A and 39 in Group B). Compared with Group B patients, Group A patients had a shorter mean anesthetic time (161.7 vs 195 minutes; p < 0.01) and operative time (71.6 vs 111 minutes; p < 0.01). The mean hospital stay was similar for the 2 groups (1.2 days for Group A vs 1.4 days for Group B; p = 0.1). Adequate postoperative data on morphological outcomes were reported by the orthotic manufacturer for 65 patients (29 in Group A and 36 in Group B). The 2 groups had similar improvement in the cephalic index (CI): Group A, mean change 10.5% (mean preoperative CI 72.6, final 80.4) at a mean follow-up of 13.2 months; Group B, mean change 12.2% (mean preoperative CI 71.0, final 79.6) at a mean follow-up of 19.4 months. The difference was not statistically significant (p = 0.15).

CONCLUSIONS

Both ESC alone and ESC with barrel staving produced excellent outcomes. However, the addition of barrel staves did not improve the results and, therefore, may not be warranted in the endoscopic treatment of sagittal craniosynostosis.

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Mark R. Proctor and Gary F. Rogers

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Alan Siu, Gary F. Rogers, John S. Myseros, Siri S. Khalsa, Robert F. Keating and Suresh N. Magge

There is no known correlation between Down syndrome and craniosynostosis. The authors report 2 infants with trisomy 21 and right unilateral coronal craniosynostosis. Both patients were clinically asymptomatic but displayed characteristic craniofacial features associated with each disorder. One patient underwent a bilateral fronto-orbital advancement and the other underwent an endoscopically assisted strip craniectomy with postoperative helmet therapy. Both patients demonstrated good cosmesis at follow-up.

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Suresh N. Magge, Arthur R. Bartolozzi IV, Neil D. Almeida, Deki Tsering, John S. Myseros, Chima O. Oluigbo, Gary F. Rogers and Robert F. Keating

OBJECTIVE

Sagittal craniosynostosis is managed with a wide variety of operative strategies. The current investigation compares the clinical outcomes of two widely performed techniques: pi craniectomy and minimally invasive endoscopic strip craniectomy (ESC) followed by helmet therapy.

METHODS

This IRB-approved retrospective study examined patients diagnosed with nonsyndromic, single-suture sagittal craniosynostosis treated with either pi craniectomy or ESC. Included patients had a minimum postoperative follow-up of 5 months.

RESULTS

Fifty-one patients met the inclusion criteria (pi 21 patients, ESC 30 patients). Compared to patients who underwent ESC, the pi patients were older at the time of surgery (mean age 5.06 vs 3.11 months). The mean follow-up time was 23.2 months for ESC patients and 31.4 months for pi patients. Initial cranial index (CI) was similar between the groups, but postoperatively the ESC patients experienced a 12.3% mean increase in CI (from 0.685 to 0.767) compared to a 5.34% increase for the pi patients (from 0.684 to 0.719), and this difference was statistically significant (p < 0.001). Median hospital length of stay (1 vs 2 days) and operative duration (69.5 vs 93.3 minutes) were significantly less for ESC (p < 0.001 for both). The ESC patients showed a trend toward better results when surgery was done at younger ages. Craniectomy width in ESC cases was positively associated with CI improvement (slope of linear regression = 0.69, p = 0.026).

CONCLUSIONS

While both techniques effectively treated sagittal craniosynostosis, ESC showed superior results compared to pi craniectomy. ESC showed a trend for better outcomes when done at younger ages, although the trend did not reach statistical significance. A wider craniectomy width (up to 2 cm) was associated with better outcomes than smaller craniectomy widths among the ESC patients.