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ABSTRACT: Congenital microstomia in the newborn can result in poor functional and aesthetic outcomes. In the past, treatment options have included surgical methods such as commissurotomies and z-plasties as well as nonsurgical treatments using oral splints. In severe microstomia, a surgical release may be required to permit splint placement. Because of the small diameter of the stoma, such surgical releases are not optimal and frequently must be repeated. We devised a nonoperative technique for the initial treatment of congenital microstomia that will slowly enlarge the stomal diameter without the need for surgical release. The patient is a newborn male born with multiple congenital anomalies including severe mandibular hypoplasia and retrusion, microstomia, and microglossia. The size of his oral cavity was less than 1 cm at birth with no clinical signs of a tongue. After initial dilation in the operating room with Hagar dilators, the oral stoma was serially dilated using Boston Scientific esophageal balloons. Once full dilation was achieved, we fabricated oral splints to continue the dilation process while maintaining oral competence with no surgical intervention.
The Cleft Palate-Craniofacial Journal 05/2011; 49(3):352-6. · 0.82 Impact Factor
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ABSTRACT: Multipotent stem cells derived from periodontal ligaments (PDLSC) and pulp of human exfoliated deciduous teeth (SHED) represent promising cell sources for bone regeneration. Recent studies have demonstrated that retinoic acid (RA) and dexamethasone (Dex) induce osteogenesis of postnatal stem cells. The objective of this study was to examine the effects of RA and Dex on the proliferation and osteogenic differentiation of SHED and PDLSC and to compare the osteogenic characteristics of SHED and PDLSC under RA treatment. SHED and PDLSC were treated with serum-free medium either alone or supplemented with RA or Dex for 21 days. The proliferation of SHED and PDLSC was significantly inhibited by both RA and Dex. RA significantly upregulated gene expression and the activity of alkaline phosphatase in SHED and PDLSC. Positive Alizarin red and von Kossa staining of calcium deposition was seen on the RA-treated SHED and PDLSC after 21 days of culture. The influences of RA on the osteogenic differentiation of SHED and PDLSC were significantly stronger than with Dex. Supplementation with insulin enhanced RA-induced osteogenic differentiation of SHED. Thus, RA is an effective inducer of osteogenic differentiation of SHED and PDLSC, whereas RA treatment in combination with insulin supplementation might be a better option for inducing osteogenic differentiation. Significantly higher cell proliferation of PDLSC results in greater calcium deposition after 3-week culture, suggesting that PDLSC is a better osteogenic stem cell source. This study provides valuable information for efficiently producing osteogenically differentiated SHED or PDLSC for in vivo bone regeneration.
Cell and Tissue Research 03/2010; 340(2):323-33. · 3.11 Impact Factor
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Mark P Mooney,
H Wolfgang Losken,
Amr M Moursi,
Jocelyn M Shand,
Gregory M Cooper,
Chris Curry,
Lillian Ho,
Anne M Burrows, Eric J Stelnicki,
Joseph E Losee,
Lynne A Opperman,
Michael I Siegel
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ABSTRACT: Postoperative resynostosis and secondary craniofacial growth abnormalities are common sequelae after craniofacial surgery. It has been suggested that an overexpression of transforming growth factor-beta2 (Tgf-beta2) may be related to craniosynostosis and contribute to postoperative resynostosis. Interference with Tgf-beta2 function using neutralizing antibodies may inhibit resynostosis and improve postoperative craniofacial growth; the present study was designed to test this hypothesis. Twenty-nine New Zealand white rabbits with bilateral coronal suture synostosis were used: 1) suturectomy controls (n=9); 2) suturectomy with nonspecific, control IgG antibody (n=9); and 3) suturectomy with anti-Tgf-beta2 antibody (n=11). At 10 days of age, a 3 mm x 15-mm coronal suturectomy was performed. The sites in groups 2 and 3 were immediately filled with 0.1 cc of a slow resorbing collagen gel mixed with either IgG (100 microg/suture) or anti-Tgf-beta2 (100 microg/suture). Three-dimensional computed tomography scan reconstructions of the skulls and cephalographs were obtained at 10, 25, 42, and 84 days of age. Computed tomography scan data revealed patent suturectomy sites and significantly (P<0.05) greater intracranial volumes by 84 days of age in rabbits treated with anti-Tgf-beta2 compared with controls. Cephalometric analysis revealed significant (P<0.05) differences in craniofacial, cranial vault, and cranial base growth by 84 days of age in rabbits treated with anti-Tgf-beta2 compared with controls. These data support the initial hypothesis that interference with Tgf-beta2 function inhibited postoperative resynostosis and improved cranial vault growth in this rabbit model. Thus, this biologically based therapy may be a potential surgical adjunct in the treatment of infants with craniosynostosis.
Journal of Craniofacial Surgery 04/2007; 18(2):336-46; discussion 347-9. · 0.82 Impact Factor
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Mark P. Mooney,
H. Wolfgang Losken,
Amr M. Moursi,
Jocelyn M. Shand,
Gregory M. Cooper,
Chris Curry,
Lillian Ho,
Anne M. Burrows, Eric J. Stelnicki,
Joseph E. Losee,
Lynne A. Opperman,
Michael I. Siegel
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ABSTRACT: Postoperative resynostosis and secondary craniofacial growth abnormalities are common sequelae after craniofacial surgery. It has been suggested that an overexpression of transforming growth factor-β2 (Tgf-β2) may be related to craniosynostosis and contribute to postoperative resynostosis. Interference with Tgf-β2 function using neutralizing antibodies may inhibit resynostosis and improve postoperative craniofacial growth; the present study was designed to test this hypothesis. Twenty-nine New Zealand white rabbits with bilateral coronal suture synostosis were used: 1) suturectomy controls (n = 9); 2) suturectomy with nonspecific, control IgG antibody (n = 9); and 3) suturectomy with anti-Tgf-β2 antibody (n = 11). At 10 days of age, a 3 mm × 15-mm coronal suturectomy was performed. The sites in groups 2 and 3 were immediately filled with 0.1 cc of a slow resorbing collagen gel mixed with either IgG (100 μg/suture) or anti-Tgf-β2 (100 μg/suture). Three-dimensional computed tomography scan reconstructions of the skulls and cephalographs were obtained at 10, 25, 42, and 84 days of age. Computed tomography scan data revealed patent suturectomy sites and significantly (P < 0.05) greater intracranial volumes by 84 days of age in rabbits treated with anti-Tgf-β2 compared with controls. Cephalometric analysis revealed significant (P < 0.05) differences in craniofacial, cranial vault, and cranial base growth by 84 days of age in rabbits treated with anti-Tgf-β2 compared with controls. These data support the initial hypothesis that interference with Tgf-β2 function inhibited postoperative resynostosis and improved cranial vault growth in this rabbit model. Thus, this biologically based therapy may be a potential surgical adjunct in the treatment of infants with craniosynostosis.
Journal of Craniofacial Surgery 02/2007; 18(2):336-346. · 0.82 Impact Factor
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ABSTRACT: OBJECTIVE: This report describes an interesting case providing a brief review of the literature and highlighting the complexity in management and definitive treatment of a premature neonate with an obstructive epignathus teratoma and polydactyly bilaterally. Additionally, this case is unique because of the extremely large size of the oral tumor in relation to the neonate's total body size.
The Cleft Palate-Craniofacial Journal 04/2006; 43(2):244-6. · 0.82 Impact Factor
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ABSTRACT: To present the first clinical use of a new bioabsorbable material, poly (D,L) lactic acid (PDLLA), in pediatric cranial-vault remodeling procedures. This discussion will highlight the benefits and detriments of PDLLA in comparison with currently used absorbable plating systems.
This was a case study documenting the first North American case in which PDLLA was used to treat craniosynostosis.
Evaluation of pure PDLLA, a copolymer product of the mixture of poly L-lactic acid and its D-isomer, was used in an 8-month-old boy with a severe phenotypic expression of sagittal craniosynostosis. No signs of elevated intracranial pressure were present, and the neurological examination did not show impairments. Total cranial-vault remodeling with the "hung-span" technique was performed. The Resorb X system, containing 2.2-mm screws and 0.6- to 1-mm-thick plates, was used to stabilize the reconstructed cranial vault.
No surgical complications occurred. The preoperative cranial index measured 62. The scaphocephalic appearance of the skull was eliminated, and the cranial index was normalized to 77. The screws and plates were less palpable than other plating systems. Twelve months postoperatively, none of the plates and screws were identifiable by external palpation.
Resorb X has been successfully used in the treatment of sagittal craniosynostosis. Its rapid rate of resorption and lower profile make it an advantageous system for pediatric skull reconstruction. This represents the first use of this product in the United States for any pathology.
The Cleft Palate-Craniofacial Journal 08/2005; 42(4):333-9. · 0.82 Impact Factor
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Plastic & Reconstructive Surgery 05/2004; 113(4):1209-17; discussion 1218. · 3.38 Impact Factor
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ABSTRACT: In contrast to adult cutaneous wound repair, early gestational fetal cutaneous wounds heal by a process of regeneration, resulting in little or no scarring. Previous studies indicate that down-regulation of HoxB13, a member of the highly conserved family of Hox transcription factors, occurs during fetal scarless wound healing. No down-regulation was noted in adult wounds. Here, we evaluate healing of adult cutaneous wounds in Hoxb13 knockout (KO) mice, hypothesizing that loss of Hoxb13 in adult skin should result in enhanced wound healing. Tensiometry was used to measure the tensile strength of incisional wounds over a 60-day time course; overall, Hoxb13 KO wounds are significantly stronger than wild-type (WT). Histological evaluation of incisional wounds shows that 7-day-old Hoxb13 KO wounds are significantly smaller and that 60-day-old Hoxb13 KO wounds exhibit a more normal collagen architecture compared with WT wounds. We also find that excisional wounds close at a faster rate in Hoxb13 KO mice. Biochemical and histochemical analyses show that Hoxb13 KO skin contains significantly elevated levels of hyaluronan. Because higher levels of hyaluronan and enhanced wound healing are characteristics of fetal skin, we conclude that loss of Hoxb13 produces a more "fetal-like" state in adult skin.
The FASEB Journal 08/2003; 17(10):1352-4. · 5.71 Impact Factor
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ABSTRACT: Fetal surgery is finding ever-increasing applications. At present, fetal surgical techniques are used only to treat diseases associated with high mortality risk or severe morbidity if left untreated prior to birth. Yet as a discipline, in utero therapy is attractive to plastic surgeons because it affords the potential to (1) provide a scarless repair, (2) correct the primary deformity, (3) prevent secondary deformities, and (4) give the parents a "normal"-appearing child at birth. Each of these potential benefits is particularly important in patients with craniofacial anomalies. This article will both review the animal models that have been used to study the potential for intrauterine plastic surgery and provide a synopsis of the benefits that fetal therapy may provide in the treatment of selected craniofacial anomalies.
World Journal of Surgery 02/2003; 27(1):108-16. · 2.36 Impact Factor
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ABSTRACT: With the modern emphasis on minimally invasive therapies, the concept of distraction is being applied in the treatment of craniosynostosis. Although specific genetic mutations have been identified in craniosynostotic patients, changes in the gene expression induced by cranial distraction have not yet been explored. The effects of cranial distraction on hedgehog and patched-1 expression were evaluated in a rabbit model for craniosynostosis. Rabbits (n = 8) were divided into four groups: affected rabbits, wild-type rabbits, affected rabbits subject to cranial distraction, and wild-type rabbits subject to distraction. Perisutural tissue was examined using immunohistochemistry in four areas: suture, endosteum, periosteum, and osteocytes, for the expression of Indian hedgehog, sonic hedgehog, and desert hedgehog and their receptor, patched-1. Two experimental groups were compared: (1) wild-type before distraction to wild-type after distraction, and (2) synostotic before distraction to synostotic after distraction. Distraction produced several variable and interesting changes in hedgehog protein presence. In wild-type rabbits, the predominant effect was a mild decrease in Indian hedgehog levels. Sonic and desert hedgehog and patched-1 protein levels were unchanged. In synostotic rabbits, the predominant effect of distraction was to decrease Indian hedgehog, sonic hedgehog, and patched-1 protein levels. This was especially true in the periosteum and endosteum. Cranial distraction of normal and affected rabbits differentially changed both the expression levels and patterns of the hedgehog and patched-1 proteins in the cranial tissues examined. These results suggest that molecular and genetic parameters of distraction and bone response may be different in craniosynostotic individuals, which may influence treatment protocols in these patients.
Plastic & Reconstructive Surgery 09/2002; 110(2):523-32. · 3.38 Impact Factor
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ABSTRACT: Craniosynostosis is characterized by premature fusion of the cranial sutures. At the molecular level, mutations in homeobox genes, transcription factors, and growth factor receptors have been implicated in the pathogenesis of this disorder, but the specific etiologic pathways have not yet been elucidated. To further study the molecular biology behind craniosynostosis, perisutural tissues in a unique rabbit model with congenital delayed-onset coronal craniosynostosis were examined for the presence of the hedgehog family of growth factors and their receptor, patched-1. Expression of desert hedgehog, Indian hedgehog, sonic hedgehog, and patched-1 was evaluated in four areas: suture, endosteum, periosteum, and osteocytes, using immuno-histochemistry (n = 8). Protein levels in affected animals were compared with protein levels in wild-type control rabbits (n = 8). Overall, sonic hedgehog, Indian hedgehog, and patched-1 protein levels were greater in affected animals. Specifically, areas of increased staining were seen along the bony interface of the endosteum and periosteum and in the osteocytes of the synostotic rabbits. Interestingly, in the suture, increased levels of Indian hedgehog and sonic hedgehog, but not patched-1, were seen. There was minimal expression of desert hedgehog in both rabbit types. The increased overall presence of hedgehog and patched-1 proteins in synostotic rabbits may be a reactive change to the disorder or part of the pathogenic process. Although the specific cause cannot be determined from the data, it is clear that the molecular milieu of the cranial sutures in synostotic rabbits is markedly different from that of wild-type rabbits.
Plastic & Reconstructive Surgery 09/2002; 110(2):515-22. · 3.38 Impact Factor
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Plastic & Reconstructive Surgery 06/2002; 109(6):2009-18. · 3.38 Impact Factor
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ABSTRACT: A long-term follow-up study of patients who underwent bilateral mandibular distraction is presented, and the results of patients with Treacher Collins syndrome and Nager syndrome are compared with results for other forms of congenital micrognathia. It was hypothesized that the factors responsible for the predetermined, syndrome-specific shape of the mandible in patients with Treacher Collins and Nager syndromes would alter the long-term results of linear (uniplanar) distraction of the mandible. Thus, over time, the mandibles would remodel to preoperative form while maintaining the increase in volume. To investigate this hypothesis, all patients treated with bilateral mandibular distraction who had at least 1.5 years of follow-up, including satisfactory cephalometric examinations, were retrospectively reviewed. Two groups were identified. Group 1 (n = 6) were Treacher Collins and Nager syndrome patients (ages, 2 to 13 years; mean, 5.2 years) and group 2 (n = 6) included other forms of bilateral, congenital micrognathia (ages, 1.5 to 19 years; mean, 8.4 years). Serial cephalometric measurements were recorded before distraction, after distraction, and at least 18 months after distraction. Mandibular mean linear distraction distance (as recorded on the device) averaged 24.5 mm in group 1 and 26.2 mm in group 2. In group 1, the antegonial angle (angle from the mandibular plane to the top of the antegonial notch) decreased after distraction by 3.8 degrees, and the antegonial notch height was reduced by 1.6 mm. The posttreatment morphologic change was modified significantly over time, with a 3.7-degree increase of the antegonial angle and a 1.2-mm deepening of the antegonial notch. In group 2, the immediate reduction in height of the antegonial notching was subtler; however, long-term recurrence of the antegonial notching was also observed. At the end of distraction, the mean group 1 gonial angle became 8 degrees more obtuse. In contrast, patients in group 2 developed a more acute angle (mean, 8 degrees). The mandibles of the Treacher Collins syndrome patients (group 1) maintained their more obtuse postdistraction gonial angle during the period of follow-up, whereas over time this change was reversed in group 2 patients. In conclusion, experience with bilateral mandibular distraction has demonstrated that long-term determination of mandibular form is more complex than either the amount of distraction or the direction of the distraction vector. The underlying genotype and the musculoskeletal milieu must be taken into account when planning distraction, as these factors tend to remodel the mandible into its preoperative shape over time, despite the fact that the increased mandibular volume and projection are maintained.
Plastic & Reconstructive Surgery 06/2002; 109(6):1819-25; discussion 1826-7. · 3.38 Impact Factor
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ABSTRACT: Costochondral grafting for reconstruction of the Pruzansky type III mandible has given variable results. Lengthening of the rib graft by means of distraction had been advocated when subsequent growth of the grafted mandible is inadequate. This retrospective study reviews a series of patients with mandibular costochondral grafts who underwent subsequent distraction osteogenesis of the graft. A retrospective review identified two patient groups: group 1 consisted of individuals (n = 9) who underwent costochondral rib grafting of the mandible followed by distraction osteogenesis several months later at a rate of 1 mm/day. Group 2 consisted of patients with Pruzansky type II mandibles who had distraction osteogenesis without prior rib grafting (n = 9). The biomechanical parameters, orthodontic treatment regimens, and complications were examined versus patient age and quality of the rib graft. Distraction osteogenesis was successfully performed in six of the rib graft patients (group 1) and in all of the group 2 individuals. On the basis of the Haminishi scale, the computed tomographic scan appearance of the regenerate was classified as "standard or external" in six of the group 1 patients and as either "agenetic" or "pillar" (fibrous union) in the remaining three patients. In group 1, the average device was expanded 23 mm (range, 20 to 30 mm). Group 2 mandibular distraction results were all classified as either standard or external, and there was an average device expansion of 22.4 mm (range, 16 to 30 mm). The length of consolidation averaged 12.6 weeks in group 1, compared with 8.5 weeks in the traditional mandibular distraction patients (group 2). The mean shift of the dental midline to the contralateral side was 2.5 mm in group 1 versus 4.0 mm in group 2. Complex multiplanar and transport distractions were successfully performed on grafts of adequate bony volume. All four patients in group 1 with tracheostomies were successfully decannulated after consolidation. Rib graft distraction complications included pin tract infections in two patients, hardware failure with premature pin pullout in one patient, and evidence of fibrous nonunions in three young patients with single, diminutive rib grafts. In group 2, there were no distraction failures. Distraction osteogenesis can be successfully performed on costochondral rib grafts of the mandible; however, the complication rate is higher than in non-rib-graft patients. Performing the technique on older, more cooperative individuals seems to reduce this risk. In addition, placement of a double rib graft or an iliac bone graft of sufficient volume to create a neomandible with greater bone stock is an absolute requirement to decrease the risk of fibrous nonunion and provide a bone base of sufficient size for retention of the distraction device and manipulation of the regenerate.
Plastic & Reconstructive Surgery 04/2002; 109(3):925-33; discussion 934-5. · 3.38 Impact Factor
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ABSTRACT: Distraction osteogenesis has evolved as a mainstream surgical technique for lengthening and augmentation of the hypoplastic mandible. As clinical experience accumulated, there developed the need to "mold" the bony regenerate to avoid the development of postdistraction malocclusion and to achieve the desired craniofacial form. Although the potential to mold the regenerate has important clinical implications, the safety and efficacy of such an acute manipulation of the bony regenerate form have not yet been investigated in the laboratory. The purpose of this study was to determine if the distraction regenerate could be molded and result in a bony union. Four adult female dogs underwent bilateral mandibular distraction with an external multiplanar device (Stryker, Osteonics). After a latency period of 5 days, the mandibles underwent linear (anteroposterior) and angular (superoinferior) distraction to produce an anterior open bite of approximately 30 degrees. At the conclusion of the distraction procedure, the distraction sites were molded to close the open bite. In two dogs, the maneuver was performed over 3 days by changing the angulation of the devices (gradual molding), and in the other two dogs, molding was achieved with a single movement (acute molding). In the latter, the distraction devices were adjusted and reapplied to allow for anatomical fixation during the consolidation period of 49 days. According to the research protocol, the mandibles were assessed serially by cephalograms and computed tomography (CT) scans. All dogs survived the study without complications. The bony regenerate was easily molded in both groups to close the surgically created open bite. After molding, all the regenerates showed CT scan evidence of solid bone (consolidation), which was classified as "extended" on the Hamanishi scale. After the dogs were killed and soft tissue was removed, the regenerate seemed to be robust on gross examination without any evidence of fibrous nonunion. In addition, histological study of the regenerate confirmed the bony union. The study demonstrates that the mandible can be successfully molded into a desired anatomical position immediately after distraction without producing a fibrous union. Furthermore, it has been demonstrated that the bony regenerate is sufficiently malleable before consolidation to undergo either acute or gradual angular molding without disturbing osteogenic potential. The ability to mold the regenerate without the fear of creating a fibrous union or destroying bony potential provides the surgeon the capability to optimize the dental occlusion and mandibular form as part of the distraction treatment process.
Journal of Craniofacial Surgery 04/2002; 13(2):205-11. · 0.82 Impact Factor
