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Malignant tumors of the external auditory canal are rare and require surgical interventions such as lateral temporal bone resection (LTBR) for localized cases. This study introduces a novel approach, the lipofilling fascia flap technique, for external auditory canal reconstruction following LTBR or modified LTBR. The technique involves augmenting the temporal fascia flap with autologous fat grafting, aiming to enhance volume and improve outcomes. Two cases are presented, demonstrating successful reconstruction with minimal complications.
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Background: Intraoperative tissue desiccation is a risk factor for infection and wound necrosis. We hypothesized that maintaining a moist environment in the surgical field would reduce perioperative complications in free flap reconstruction for head and neck cancer surgery. Methods: A total of 331 patients who underwent free flap reconstruction after head and neck cancer surgery from January 2013 to December 2017 at Osaka International Cancer Institute were included in the study. We classified patients into two groups: those in whom saline was sprayed only during reconstruction (control group: 162 patients) and those in whom saline was sprayed in the surgical field intermittently during cancer resection and reconstruction (moisturized group: 169 patients). We compared perioperative complications, including intraoperative arterial anastomotic thrombosis, between the two groups. Other candidate risk factors for intraoperative arterial thrombosis that were assessed included a history of preoperative irradiation, history of neck surgery, advanced age, and flap type. Results: Rates of abscess formation and intraoperative arterial thrombosis were significantly lower in the moisturized group than in the control group (6.5% versus 12.7%, P < 0.05 and 3.0% versus 11.1%, P < 0.01, respectively). Binomial logistic regression analysis revealed that, in addition to no moisturization during cancer resection, advanced age and jejunal flap use were significant risk factors for intraoperative arterial thrombosis. Conclusions: Moisturization of the surgical field during cancer resection reduced the rate of perioperative complications in free flap reconstruction. Although simple, preventing desiccation of the surgical field by spraying saline solution intermittently had significant benefits on postoperative complications.
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In cutaneous T-cell lymphoma (CTCL), which arises from skin-tropic memory T cells, malignant T cells and benign T cells are confined in the same skin lesions. It is thus difficult to evaluate the phenotypic characteristics and functional activities of benign T cells in CTCL. Disialoganglioside with three glycosyl groups (GD3) is increasingly expressed on the surface of solid malignant tumor cells and takes part in tumor progression and suppression of tumor immunity. However, the role of GD3 in CTCL is not well-understood. In this study, the malignant and benign T cells in CTCL skin lesions were distinguished by flow cytometry and their phenotypic characteristics were compared with those of T cells from control skin specimens. In CTCL skin lesions, the benign T cells included limited resident memory T cells (TRM), which are sessile in skin and known to exert strong antitumor function. The benign T cells showed diminished Th17 property, and the expression of GD3 was high in the malignant T cells. The expression of GD3 in the malignant T cells inversely correlated with IL-17A production from the benign CD4 T cells. GD3 from the malignant T cells was implied to be involved in suppressing the Th17 activity of the benign T cells independent of the regulation of TRM differentiation in CTCL. Revealing the role of GD3 in inhibiting the production of IL-17A in CTCL would aid the understanding of the suppressive mechanism of the antitumor activity by malignant tumor cells.
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Gangliosídeos/metabolismo , Linfoma Cutâneo de Células T/imunologia , Neoplasias Cutâneas/imunologia , Pele/patologia , Células Th17/imunologia , Adulto , Idoso , Idoso de 80 Anos ou mais , Biópsia , Separação Celular , Feminino , Citometria de Fluxo , Humanos , Linfoma Cutâneo de Células T/patologia , Masculino , Pessoa de Meia-Idade , Pele/imunologia , Neoplasias Cutâneas/patologia , Células Th17/metabolismoRESUMO
BACKGROUND: Latissimus dorsi myocutaneous (LD) flaps are widely used in breast reconstruction surgery. However, seromas often form postoperatively at the donor site as a complication. This study aimed to determine the impact of different electrocautery modes during flap elevation, with or without subsequent quilting sutures, on postoperative seroma formation. METHODS: Subjects were 112 patients who underwent immediate breast reconstruction with LD flaps after breast-conserving surgery between April 2015 and January 2019. Group A consisted of 21 patients who underwent LD flap elevation using the fulgurate mode, and group B consisted of 25 patients who underwent flap elevation using the lower power, pure cut mode. Group C consisted of 66 patients who underwent flap elevation with the lower power, pure cut mode combined with quilting sutures for wound closure. RESULTS: Mean 1-week postoperative back drain volume and the mean number of days to drain removal in group B were significantly reduced relative to those in group A (group A, 752.3 mL and 16.9 days, respectively; group B, 552.2 mL and 10.6 days, respectively; P < 0.001 for both). Group C, which included quilting sutures, had even lower values than group B (459.7 mL and 7.4 days, respectively; group B vs group C: P = 0.03 and P < 0.001, respectively). Significant differences were observed between groups for postoperative seroma formation at the flap donor site (group A, 16 [76.2%] of 21 patients; group B, 11 [44%] 25 patients; group C, 4 [6.1%] of 66 patients; A vs B: P < 0.001, B vs C: P < 0.001). CONCLUSIONS: The use of low power, pure cut mode for LD flap harvest, combined with quilting sutures, effectively shortened the time to drain removal and suppressed seroma formation. Given that specialized devices and materials are not required, this combination may reduce both patient burden and medical costs.
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Mamoplastia , Retalho Miocutâneo , Músculos Superficiais do Dorso , Eletrocirurgia , Humanos , Complicações Pós-Operatórias/etiologia , Complicações Pós-Operatórias/prevenção & controle , Seroma/etiologia , Seroma/prevenção & controle , Músculos Superficiais do Dorso/transplante , Técnicas de Sutura , SuturasRESUMO
Immediate fat grafting to the latissimus dorsi myocutaneous (LD) flaps is a breakthrough that addresses the issue of insufficient volume of LD. However, the use of this procedure in Asian patients has not yet been reported. Retrospective chart reviews were conducted on 54 Japanese cases of total breast reconstruction using fat-augmented LD flaps at our hospital from September 2017 to June 2019. There were 24 immediate reconstruction cases, 18 immediate two-stage reconstruction cases, nine delayed reconstruction cases, and three delayed two-stage reconstruction cases. Median age was 46 years (range, 29-69 years), and median body mass index was 21.5 (17-33.8). Median mastectomy specimen and flap weight was 225â¯g (123-993) and 225â¯g (130-796), respectively. The median volume of fat graft was 114â¯ml (46-305) for the LD flap and 58â¯ml (15-200) for the pectoralis major muscle. Of the 53 completed reconstruction cases, 38 (71.7%) achieved sufficient volume with the initial operation and six (11.3%) required additional fat grafting. The proportion of cases in the immediate reconstruction group, which achieved sufficient volume in the initial operation was significantly higher than those of the other three reconstruction groups (pâ¯=â¯0.007). Total breast reconstruction with fat-augmented LD flaps is a viable procedure for thin patients who have insufficient abdominal tissue, for those who wish to avoid abdominal scars, and for those in whom abdominal flaps have already been used. The procedure allows for large volume transplantation even with small skin paddles, which allows for smaller skin paddles to be designed without the need for extensive subcutaneous dissection.
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Tecido Adiposo/transplante , Neoplasias da Mama , Mamoplastia , Mastectomia , Retalho Miocutâneo/transplante , Músculos Peitorais/transplante , Complicações Pós-Operatórias , Músculos Superficiais do Dorso/transplante , Neoplasias da Mama/epidemiologia , Neoplasias da Mama/cirurgia , Feminino , Humanos , Japão/epidemiologia , Mamoplastia/efeitos adversos , Mamoplastia/métodos , Mastectomia/efeitos adversos , Mastectomia/métodos , Pessoa de Meia-Idade , Avaliação de Processos e Resultados em Cuidados de Saúde , Complicações Pós-Operatórias/diagnóstico , Complicações Pós-Operatórias/etiologia , Estudos RetrospectivosRESUMO
Latissimus dorsi (LD) flaps are widely used in breast reconstruction for their ease of use and minimal sacrifice of the donor site. Various strategies to increase flap volume have been suggested, but tissue volume is often insufficient for patients with little subcutaneous fat. While lumbar artery perforator (LAP) flaps are advantageous for thin patients because they allow for the addition of a large amount of fat, the vascular pedicle is short and vascular grafts are often required. To address these shortcomings, we propose here a LD-LAP chimeric flap. Specifically, the LD flap and LAP flap are elevated as one piece, and the 6th or 7th intercostal artery perforators and lumbar artery perforators, which are harvested together with the flap, are connected via intra-flap crossover anastomosis. Anastomosis for both intercostal artery perforators and lumbar artery perforators was performed about 1 cm away from the flap. Indocyanine green angiography performed after anastomosis showed improved blood flow to the LAP flap portion of the chimeric flap. The chimeric flap was used in 4 patients, with a mean flap volume of 460 ml (range, 300-690 ml) and mean duration of 439 minutes (393-484 minutes) for reconstruction surgery. During the mean follow-up period of 29.5 months (range, 16-40 months), sufficient tissue volume was obtained and none of the patients developed flap necrosis. Although our method requires vascular anastomosis and may extend operative time, it substantially increases LD flap volume and thus is likely to be an effective auxiliary component to breast reconstruction using LD flaps.
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BACKGROUND: In the typical procedure for secondary correction of the inframammary fold (IMF) following breast reconstruction, a large incision is often required, and this increases surgical invasiveness. The "drawstring method" is a simple procedure for recreating a smooth IMF. We modified the drawstring method and developed an essentially scarless method for IMF correction from small stab incisions. METHODS: Patients at our hospital who presented with IMF ptosis or loss of definition after breast reconstruction and required IMF correction, as well as those who requested IMF recreation for the contralateral breast, during the period spanning May 2016 to June 2019 were considered for this study. We collected and analyzed demographic data, as well as complications and postoperative outcomes. RESULTS: The new method was performed on 20 patients, with the following breakdown: IMF recreation after breast reconstruction with a deep inferior epigastric artery perforator flap (11 patients), IMF recreation after breast reconstruction with a breast implant (2 patients), IMF recreation after breast reconstruction with fat graft (5 patients), and IMF recreation for the contralateral breast (2 patients). Overcorrection of the IMF stabilized by 2-3 months postoperatively, resulting in a smooth and well-defined IMF. For non-breast implant cases, the implant volume increased at the lower pole. Slack in the suture was observed in only 2 patients of the deep inferior epigastric artery perforator group and in 1 patient of the breast implant group after 6 months postoperatively. CONCLUSIONS: Our new method allows for the recreation of an essentially scarless, smooth, and well-defined IMF. IMF definition can be adjusted by altering the depth of the barbed suture. Since this method can be performed under local anesthesia, it offers the benefits of reducing medical costs and physical burden on patients.
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While the use of free flaps has become routine and is associated with a low complication rate, pedicled flaps remain a solid reconstructive option in various clinical situations. Pedicled flaps provide a reliable vascular supply and involve a simple surgical procedure. Although the procedure is advantageous from the standpoint of a low rate of flap ischemia, thrombosis, and total flap loss, these complications are still occasionally observed due to intraoperative pedicle injury, postoperative torsion, or compression. Here we report on a case of severe venous thrombosis in a pedicled latissimus dorsi (LD) flap used for breast reconstruction. The patient was a 52-year-old woman who underwent mastectomy and immediate breast reconstruction with a LD flap for left breast cancer. Postoperatively, the color of the skin paddle became dark blue over time. Emergent surgical exploration revealed kinking and narrowing of the thoracodorsal vessels and extensive venous thrombi. The kinked pedicles were repaired and selective thrombolytic therapy was performed. A thrombolytic agent was administered through the serratus anterior branch of the thoracodorsal artery in retrograde fashion while the thoracodorsal vessels were clamped just cephalad to the bifurcation. This allowed for draining of the thrombolytic agent and thrombi through the serratus anterior branch of the thoracodorsal vein without flowing into the systemic circulation. To the best of our knowledge, this is the first report of selective thrombolysis using a pedicle branch to treat venous thrombosis in a pedicled flap. If major vascular branches are available in a pedicled flap, selective thrombolytic therapy may be possible without disconnecting the pedicle, as in the present case.
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BACKGROUND: "Window" resection of the trachea is sometimes performed to remove tumors invading the trachea. Here, we present a novel reconstructive procedure to this end. METHODS: Eleven patients (mean age, 64 years; range, 46-80 years) were included. Primary diagnoses included thyroid cancer and adenoid cystic carcinoma of the trachea. All defects were partial and located in the neck (mean width and length, 3/5 circle and 7.5 rings; range, 1/2-2/3 circle and 5-9 rings). Immediate 2-stage reconstruction was performed using a forearm flap and free bone graft. The bone graft was utilized as a supportive skeleton. A tracheostoma was left open for several months following the initial surgery, and then closed. RESULTS: The mean flap size was 6.1 × 9.7 cm (range, 6-7 × 7-16 cm). Mean number of grafted bone strips and length were 1.6 (range, 1-3) and 6.1 cm (range, 4.5-7 cm). All flaps survived. Five patients developed complications in the neck, including surgical site infections (SSIs), recurrent nerve palsy, and lymphorrhea. Four patients developed donor site complications, including clavicular fracture and SSIs. Mean postoperative follow-up lasted 85 months (range, 11-149 months). Normal speech was restored in 9 patients. Stoma closure was abandoned in 2 patients, because 1 patient showed vocal cord fixation with advanced age and the other showed bone graft loss following SSI. CONCLUSIONS: Creating a tracheostoma during the first operation prevents postoperative airway compromise. Our bone graft placement easily achieves tracheal rigidity. This procedure is simple and safe for tracheal window defect repair.
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Transplante Ósseo/métodos , Procedimentos de Cirurgia Plástica/métodos , Retalhos Cirúrgicos , Neoplasias da Traqueia/cirurgia , Idoso , Idoso de 80 Anos ou mais , Feminino , Antebraço , Humanos , Masculino , Pessoa de Meia-Idade , Estudos Retrospectivos , TraqueostomiaRESUMO
BACKGROUND: When pharyngoesophagectomy is performed in conjunction with anterior mediastinal tracheostomy, reconstructing both the trachea and alimentary tract is extremely difficult. We developed a novel 1-stage reconstructive procedure using a single free jejunal flap containing multiple vascular pedicles to decrease postoperative morbidity and mortality. Free jejunal flap transfer with multiple vascular pedicles could offer a viable option for reducing associated life-threatening complications. METHODS: We performed a retrospective review of 34 patients who underwent free jejunal flap transfer with multiple vascular pedicles in anterior mediastinal tracheostomy and pharyngoesophagectomy due to lesions involving both the airway and esophagus. In all cases, 1-stage reconstruction of the digestive tract and trachea was performed. Technical details and outcomes were analyzed. RESULTS: All 34 jejunal flaps (100%) survived. Major morbidity classified as Clavien-Dindo grades III and IV occurred in 10 (29.4%) and 0 (0%) patients, respectively during hospitalization. With regard to common complications, anastomotic leakage from transferred jejunal flaps and surgical site infections occurred in 0 (0%) and 7 (20.6%) patients, respectively. Five (14.7%) patients experienced tracheal stoma dehiscence. Donor site morbidity was observed in 2 (5.9%) patients. The overall in-hospital mortality rate was 2.9%. CONCLUSIONS: Our 1-stage reconstruction procedure achieved low morbidity and low mortality rates following anterior mediastinal tracheostomy and pharyngoesophagectomy. Only 1 jejunal flap transfer is needed to simultaneously reconstruct the trachea and alimentary tract in a safe and reliable manner with this procedure.
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Esofagectomia , Retalhos de Tecido Biológico , Esvaziamento Cervical , Faringectomia , Procedimentos de Cirurgia Plástica/métodos , Traqueostomia , Idoso , Neoplasias Esofágicas/patologia , Neoplasias Esofágicas/cirurgia , Feminino , Humanos , Neoplasias Hipofaríngeas/patologia , Neoplasias Hipofaríngeas/cirurgia , Masculino , Pessoa de Meia-Idade , Estudos RetrospectivosRESUMO
BACKGROUND: In pharyngoesophageal reconstruction, we transferred a long jejunum flap containing multiple pedicles to achieve a 100% flap survival rate, and used the redundant mesentery to cover important tissues and fill in the dead space to reduce common postoperative complications, such as surgical site infections and fistula formation. METHODS: A total of 243 jejunal flap transfers with multiple vascular anastomoses were reviewed to examine flap survival and rates of early postoperative complications, such as surgical site infections and fistula formation, perioperative mortality, and donor site morbidity. RESULTS: All 243 jejunal flaps survived without any partial necrosis. The surgical site infections occurred in 15 cases (6.2%) and fistula formation in 9 cases (3.7%). The perioperative mortality rate was 0.4%. There were 7 cases (2.9%) with donor site morbidity. CONCLUSION: Although our procedure requires extra operating time for additional vessel anastomoses, it could be performed safely and reliably with a high success rate.
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Neoplasias Esofágicas/cirurgia , Retalhos de Tecido Biológico/irrigação sanguínea , Jejuno/transplante , Neoplasias Faríngeas/cirurgia , Adulto , Idoso , Idoso de 80 Anos ou mais , Fístula Cutânea/etiologia , Esofagectomia , Feminino , Sobrevivência de Enxerto , Humanos , Masculino , Pessoa de Meia-Idade , Faringectomia , Complicações Pós-Operatórias , Estudos Retrospectivos , Infecção da Ferida Cirúrgica/etiologiaRESUMO
OBJECTIVE: We invented a two-tiered structure device based on stereolithography for residual mandible repositioning in mandibular reconstruction with fibular flap, and examined its usefulness. PATIENTS AND METHODS: A total of eight patients (six carcinomas and two osteomyelitis) who had undergone mandibular reconstruction with fibular flap were included. Mandibular defects according to Jewer's classification were L for five, LC for two, and H for one patient (range of bone defect size, 7.7-13.3 cm). Based on a stereolithographic mandibular model, a two-tiered structure device was made preoperatively with resin, and was applied during surgery to define the accurate position of residual mandible following segmental mandibulectomy. Postoperative aesthetic and functional outcomes, including dental status, diet, and speech were evaluated. RESULTS: The device was applied without any problems during surgery. Follow-up period ranged from eight to twenty-two months. Good postoperative alignment of the grafts and occlusion were also achieved in all eight patients. Five patients were able to return to eating meals as they had pre-surgery, and two patients required dental rehabilitation with dentures. As for speech, four patients scored ten points, three patients scored eight points, and one patient scored four points in Hirose's scoring system, which means that seven patients was rated as excellent, and one patient was rated as poor. Aesthetic outcomes were excellent in three patients, good in three patients, and fair in two patients. CONCLUSION: Our device was easy to prepare, successfully maintained the precise position of the residual mandible, and facilitated bone graft cutting and insetting during reconstruction. © 2016 Wiley Periodicals, Inc. Microsurgery 37:509-515, 2017.
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Transplante Ósseo/métodos , Fíbula/cirurgia , Neoplasias Mandibulares/cirurgia , Reconstrução Mandibular/métodos , Microcirurgia/instrumentação , Idoso , Idoso de 80 Anos ou mais , Estudos de Coortes , Feminino , Fíbula/transplante , Seguimentos , Humanos , Cuidados Intraoperatórios/métodos , Masculino , Mandíbula/cirurgia , Neoplasias Mandibulares/diagnóstico , Reconstrução Mandibular/instrumentação , Pessoa de Meia-Idade , Recuperação de Função Fisiológica/fisiologia , Estudos Retrospectivos , Estereolitografia , Dispositivos de Fixação Cirúrgica , Resultado do TratamentoRESUMO
Recent advances in perforator flap surgical techniques have allowed for safe and reliable autologous tissue transfer with minimal donor-site morbidity. Between April 2012 and January 2013, we performed immediate breast reconstruction using free medial circumflex femoral artery perforator (MCFAP) flaps in 15 patients after breast-conserving surgery. The flaps were harvested from patients in the lithotomy position, while a second surgical team simultaneously conducted tumour resection. Of the 15 procedures performed, three flaps were dissected as true perforator flaps, while 12 flaps were dissected as muscle-sparing perforator flaps. The mean flap weight was 138.5 g (range, 77-230 g) and the mean pedicle length was 5.7 cm (range, 3.0-9.0 cm). Recipient vessels for anastomosis were serratus branches in 10 patients, internal mammary vessels in three patients and thoracodorsal vessels in two patients. The mean duration of surgery was 6.74 h (range, 5.65-9.45 h). There were no major complications requiring surgical intervention. Researchers observed partial flap necrosis, which manifested as small firm lesions in two patients, as well as local wound infection and dehiscence in one patient, which resolved spontaneously. There were no instances of donor-site seroma formation or lymphoedema in any of the patients. An objective assessment of postoperative photographs showed that cosmetic results were mostly satisfactory. Donor-site scars along the medial groin crease were inconspicuous and readily concealed by clothing. Given its reliable vascularity and minimal donor-site morbidity, the free MCFAP flap can be a good alternative for partial breast reconstruction after breast-conserving surgery.
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Neoplasias da Mama/cirurgia , Mamoplastia/métodos , Retalho Perfurante , Sítio Doador de Transplante/cirurgia , Adulto , Idoso , Feminino , Virilha/cirurgia , Humanos , Mamoplastia/efeitos adversos , Mastectomia Segmentar , Pessoa de Meia-Idade , Duração da Cirurgia , Retalho Perfurante/efeitos adversos , Retalho Perfurante/irrigação sanguínea , Fatores de Tempo , Sítio Doador de Transplante/irrigação sanguíneaRESUMO
NELL1 is a secretory osteogenic protein containing several structural motifs that suggest that it functions as an extracellular matrix component. To determine the mechanisms underlying NELL1-induced osteoblast differentiation, we examined the cell-adhesive activity of NELL1 using a series of recombinant NELL1 proteins. We demonstrated that NELL1 promoted osteoblastic cell adhesion through at least three cell-binding domains located in the C-terminal region of NELL1. Adhesion of cells to NELL1 was strongly inhibited by function-blocking antibodies against integrin α3 and ß1 subunits, suggesting that osteoblastic cells adhered to NELL1 through integrin α3ß1. Further, focal adhesion kinase activation is involved in NELL1 signaling.
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Proteínas de Ligação ao Cálcio/química , Regulação da Expressão Gênica , Glicoproteínas/química , Integrina alfa3beta1/biossíntese , Osteoblastos/citologia , Células 3T3 , Animais , Proteínas de Ligação ao Cálcio/fisiologia , Adesão Celular , Linhagem Celular Tumoral , Relação Dose-Resposta a Droga , Glicoproteínas/fisiologia , Humanos , Sistema de Sinalização das MAP Quinases , Camundongos , Camundongos Endogâmicos C3H , Modelos Biológicos , Plasmídeos/metabolismo , Estrutura Terciária de Proteína , Proteínas Recombinantes/química , Transdução de SinaisRESUMO
NELL1 is a secretory protein that induces osteogenic differentiation and bone formation by osteoblastic cells. Because of its potent osteoinductive activity, NELL1 may be useful for bone regeneration therapy. However, at present, we have little knowledge regarding NELL1 receptors and NELL1-mediated signaling pathways. We have previously produced NELL1 using an insect's cell expression system; however, the protein was relatively unstable and was degraded by proteases released from dead cells. In the present study, NELL1 protein was expressed in human embryonic kidney 293-F cells. Stable cell lines expressing NELL1 fused to a C-terminal hexahistidine-tag were obtained by G418 selection of transfected cells. Cells grown in serum-free medium showed high levels of NELL1 protein production (approximately 4 mg/l cell culture) for up to 6 months. NELL1 protein was purified from culture medium using a one-step nickel-chelate affinity chromatography protocol. Purified NELL1 protein immobilized onto culture dishes induced the expression of both early and late osteogenic markers on mouse mesenchymal C3H10T1/2 cells. When NELL1-expressing 293-F cells were grown on gelatin-coated glass cover slips, recombinant NELL1 was deposited in the extracellular matrix after detachment of cells. These results suggest that NELL1 acts as an extracellular matrix component. Recombinant NELL1 formed multimers and was glycosylated. An abundant source of functionally active NELL1 protein will be useful for more advanced studies, such as the development of novel techniques for bone regeneration.
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Biotecnologia/métodos , Proteínas do Tecido Nervoso/biossíntese , Proteínas Recombinantes/biossíntese , Animais , Biomarcadores/metabolismo , Proteínas de Ligação ao Cálcio , Separação Celular , Cromatografia de Afinidade , Eletroforese em Gel de Poliacrilamida , Matriz Extracelular/metabolismo , Vetores Genéticos/genética , Glicosilação , Células HEK293 , Humanos , Camundongos , Proteínas do Tecido Nervoso/isolamento & purificação , Osteogênese , Processamento de Proteína Pós-Traducional , Proteínas Recombinantes/isolamento & purificação , Fatores de TempoRESUMO
PURPOSE: This study reports the senior author's experience of orthognathic surgery in patients with prognathism and undiagnosed type I osteogenesis imperfecta and includes a review of the literature. PATIENTS AND METHODS: Two patients with undiagnosed type I osteogenesis imperfecta underwent orthognathic surgery for correction of prognathism at Chang Gung Craniofacial Center, Taipei, Taiwan. The initial surgical plan was to perform 2-jaw orthognathic surgery in both patients. RESULTS: The bone quality was found to be fragile during the operation, and the original plan was changed intraoperatively to 1-jaw mandibular surgery. Both operations were performed without complications, and wound healing progressed normally. Both the final facial profile and occlusal outcome were satisfactory in 1 patient, with mild relapse occurring in the second patient. CONCLUSIONS: For patients with type I osteogenesis imperfecta, the orthognathic surgery plan should be simplified as much as possible. Prolonged intermaxillary fixation is recommended to facilitate bone union. Complications could possibly be avoided.