Lipoprotein Apheresis Alleviates Treatment-Resistant Peripheral Artery Disease Despite the Normal Range of Atherogenic Lipoproteins: The LETS-PAD Study.

AIM: Peripheral artery disease (PAD) severely impairs patient prognosis and quality of life (QOL). Although lipoprotein apheresis (LA) has been applied to patients with PAD and elevated serum atherogenic lipoproteins, we hypothesized that LA can be effective for treating PAD even in patients with controlled serum lipoproteins through pleiotropic anti-atherosclerotic effects beyond lipoprotein removal. This study aimed to evaluate the efficacy of LA in patients with treatment-resistant PAD and controlled serum lipoproteins focusing on QOL. METHODS: In a single-arm prospective study, 30 patients with refractory PAD who had controlled serum lipoproteins underwent sequential LA sessions using dextran sulfate adsorption columns, aiming to complete 10 sessions. The ankle-brachial pressure index (ABI) and vascular QOL (VascuQOL) score were evaluated as the primary outcomes. Secondary outcomes included reactive hyperemia index (RHI) and biological antioxidant potential (BAP) as an endothelial function test and serum antioxidative-capacity evaluation, respectively. RESULTS: ABI significantly increased after LA sessions (pre-treatment 0.60±0.09 vs. post-treatment 0.65±0.13, p=0.023). Total VascuQOL score (3.7±1.1 vs 4.6±1.1, p＜0.001) and RHI (1.70±0.74 vs 2.34±1.76, p=0.023) significantly improved after the LA sessions. BAP tended to increase after the LA sessions, and the change reached statistical significance 3 months after treatment. CONCLUSION: ABI and QOL improved after a series of LA sessions in conventional treatment-resistant PAD patients with controlled serum lipoprotein levels. Increased antioxidative capacity and ameliorated endothelial function were observed after the LA treatment.

Submucosal dissection to close wide cleft palate with folded mucoperiosteum for bilateral cleft lip and palate with popliteal pterygium syndrome.

Characteristic features of popliteal pterygium syndrome (PPS) associated with the craniofacial region include cleft palate, syngnathia, and difficulty with reconstruction. We developed a new procedure of submucosal dissection with periosteotomy to close the folded mucosa in bilateral cleft lip and palate patients with PPS. This technique could be applicable for patients with wide cleft palate.

Pectoralis Major Myocutaneous Flap Revisited as a Workhorse for Reconstruction for Defects in the Upper Arm: A Case Report.

Many previous reviews of the literature have described the grafts and techniques for management of defects in the upper arm. However, the alternatives are limited in cases where some conventional flaps are not available and the nearby donor vessels have been previously sacrificed for free flaps. A 77-year-old man presented with a tumor in the right upper arm just above the axilla. The patient had already undergone surgeries for three recurrences of low-grade myxofibrosarcoma, the primary site of which was around the right scapula. The pectoralis major musculocutaneous flap was used for the defect caused by tumor resection, since there was no other available option. An acceptable result was obtained without any major complications. Thus, the pectoralis major myocutaneous flap may be a candidate for reconstruction of defects in the proximal part of the upper arm.

A predictor of a postoperative fistula after double-opposing Z-plasty in bilateral cleft lip and palate patients.

Furlow double-opposing Z-plasty (FDOZ) as primary palatoplasty for the bilateral cleft lip and palate (BCLP) closure often leads to a palatal fistula formation in cases with wide clefts. We examined the utility of the calculated maximum closable cleft width to determine the feasibility of a direct palatal closure without lateral incisions. In this retrospective study, we analyzed consecutive patients with a BCLP who were treated for 5 years from 2009. In sixty-three BCLP patients, the following dimensions were measured preoperatively: an actual distance between maxillary tuberosities (Actual X); actual cleft height, calculated from the line joining the maxillary tuberosities to the cleft edge (Actual Y); and actual cleft width at the hard-soft palate junction (Actual Z) and calculated maximum cleft width requiring a direct closure (CMZ). Six months postoperatively, the relationships between the fistula occurrence and actual Z/CMZ were examined. Median values (interquartile range) of Actual X, Y, and Z and CMZ were 35.0 (33.0-39.0) mm, 7.0 (6.0-8.0) mm, 10.0 (8.0-11.0) mm, and 4.6 (2.7-5.7) mm, respectively. The median age at operation was 8.0 (7.0-17.0) months. Fistulae at the hard-soft palate junction developed postoperatively in three cases with more than a 10-mm cleft width (4.76%). A receiver operating characteristic (ROC) curve analysis indicated that actual Z was a better predictor of a postoperative fistula formation than CMZ. An actual cleft width was a better predictor of the fistula occurrence than a calculated maximum cleft width after FDOZ for a BCLP repair. The low fistula rate of FDOZ suggested that FDOZ could be used to close the palate with less than a 10-mm cleft width.

Posterior pharyngeal wall augmentation using autologous tiered costal cartilage for velopharyngeal insufficiency.

The aim of this study was to retrospectively estimate intervention outcomes and to establish suitable indications for autogenous tiered cartilage augmentation (ATCA) of the posterior pharyngeal wall for velopharyngeal insufficiency (VPI). The sample cohort comprised 17 consecutive patients with VPI (10 boys, 7 girls), excluding syndromic cases. Pre- and postoperative findings were comprehensively evaluated using a combination of speech assessment and lateral pharyngography (LPG). Hypernasality and consonant distortion were scored (normal: 0; mild: 1; moderate: 2; severe: 3). Velopharyngeal gap size at rest (AC) and at maximum closure (BC) were measured on LPG. All patients displayed improvements in velopharyngeal function and speech score, and no patients required re-operation. Pre- and postoperative hypernasality scores were 1.9 ± 0.5 and 0.6 ± 0.5, respectively (P < .001). Pre- and postoperative consonant distortion scores were 1.9 ± 0.6 and 0.6 ± 0.5, respectively (P < .001). Pre- and postoperative AC distances were 8.2 ± 3.4 mm and 5.9 ± 2.6 mm, respectively (P < .001). Pre- and postoperative BC distances were 3.9 ± 2.4 mm and 0.3 ± 0.8 mm, respectively (P < .001). Conclusions: ATCA appears effective for surgical treatment of VPI patients with a gap less than 10 mm.

A phase III, multicenter, single-arm study to assess the utility of indocyanine green fluorescent lymphography in the treatment of secondary lymphedema.

OBJECTIVE: Indocyanine green (ICG) fluorescent lymphography might be useful for assessing patients undergoing lymphatic surgery for secondary lymphedema. The present clinical trial aimed to confirm whether ICG fluorescent lymphography would be useful in evaluating lymphedema, identifying lymphatic vessels suitable for anastomosis, and confirming patency of lymphaticovenular anastomosis in patients with secondary lymphedema. METHODS: The present phase III, multicenter, single-arm, open-label, clinical trial (HAMAMATSU-ICG study) investigated the accuracy of lymphedema diagnosis via ICG fluorescent lymphography compared with lymphoscintigraphy, rate of identification of lymphatic vessels at the incision site, and efficacy for confirming patency of lymphaticovenular anastomosis. The external diameter of the identified lymphatic vessels and the distance from the skin surface to the lymphatic vessels using preoperative ICG fluorescent lymphography were measured intraoperatively under surgical microscopy. RESULTS: When the clinical decision for surgery at each research site was made, the standard diagnosis of lymphedema was considered correct. For the 26 upper extremities, a central judgment committee who was unaware of the clinical presentation confirmed the imaging diagnosis was accurate for 100.0% of cases, whether the assessments had been performed via lymphoscintigraphy or ICG lymphography. In contrast, for the 88 lower extremities, the accuracy of the diagnosis compared with the diagnosis by the central judgment committee was 70.5% and 88.2% for lymphoscintigraphy and ICG lymphography, respectively. The external diameter of the identified lymphatic vessels was significantly greater in the lower extremities than in the upper extremities (0.54 ± 0.21 mm vs 0.42 ± 0.14 mm; P < .0001). Also, the distance from the skin surface to the lymphatic vessels was significantly longer in the lower extremities than in the upper extremities (5.8 ± 3.5 mm vs 4.4 ± 2.6 mm; P = .01). For 263 skin incisions, with the site placement determined using ICG fluorescent lymphography, the rate of identification of lymphatics vessels suitable for anastomosis was 97.7% (95% confidence interval, 95.1%-99.2%). A total of 267 lymphaticovenular anastomoses were performed. ICG fluorescent lymphography was judged as "useful" for confirming patency after the anastomosis in 95.1% of the cases. CONCLUSIONS: ICG fluorescent lymphography could be useful for improving the treatment of patients with secondary lymphedema from the outpatient setting to surgery.

Pathological Changes in the Lymphatic System of Patients with Secondary Lower Limb Lymphedema Based on Single Photon-Emission Computed Tomography/Computed Tomography/Lymphoscintigraphy Images.

Background: In patients with secondary upper limb lymphedema, positive correlations have been observed between the dermal back flow (DBF) type and visualization of lymph nodes around the clavicle, between the former and the lymph flow pathway type, and between the latter and the visualization of lymph nodes around the clavicle when using single photon-emission computed tomography/computed tomography/lymphoscintigraphy (SPECT-CT LSG). Methods and Results: We analyzed the associations between the visualization of inguinal lymph nodes, the lymph flow pathway type, and the DBF type using SPECT-CT LSG in 81 patients with unilateral secondary lower limb lymphedema by statistical analysis using Fisher's exact test. We revealed that the lymph flow pathways in the lower limb can be classified into nine types because the type in the lower leg is not always equal to the type in the thigh. Associations were observed between the visualization of inguinal lymph nodes and types of DBF (p < 0.01), between the types of lymph flow pathway in the thighs and visualization of the inguinal lymph nodes (p = 0.02), and between the lymph flow pathway types in the thighs and lower legs (p < 0.01). Conclusion: Detriment to the superficial lymph flow pathways in the lower limb appears to usually start from the proximal side, and deep pathways are considered to become dominant from a compensatory perspective as lymphedema severity increases.

A procedure combining double opposing Z-plasty with buccal flap and skin graft for a cleft palate patient with short palate.

Cleft palate patients with a short palate are sometimes encountered and it is difficult to achieve effective primary palatoplasty and good speech in these cases. Our purpose was to establish an effective palatoplasty for a cleft palate patient with Randall type III short palate. Buccal musculomucosal flap on the nasal side and skin graft on the oral side were performed, along with double opposing Z-plasty. Speech improved postoperation. This procedure brought the nasopharyngeal area closer to the normal anatomical state. In terms of disadvantages, the procedure is rather complicated and depends on the engraftment rate.

Precision modeling of gall bladder cancer patients in mice based on orthotopic implantation of organoid-derived tumor buds.

Genetically engineered mice (GEM) are the gold standard for cancer modeling. However, strict recapitulation of stepwise carcinogenesis from a single tumor-initiating epithelial cell among genetically intact cells in adults is not feasible with the currently available techniques using GEM. In previous studies, we partially overcame this challenge by physically isolating organs from adult animals, followed by genetic engineering in organoids and subcutaneous inoculation in nude mice. Despite the establishment of suitable ex vivo carcinogenesis models for diverse tissues, tumor development remained ectopic and occurred under immunodeficient conditions. Further refinement was, therefore, necessary to establish ideal models. Given the poor prognosis and few models owing to the lack of gall bladder (GB)-specific Cre strain, we assumed that the development of authentic models would considerably benefit GB cancer research. Here, we established a novel model using GB organoids with mutant Kras and Trp53 loss generated in vitro by lentiviral Cre transduction and CRISPR/Cas9 gene editing, respectively. Organoid-derived subcutaneous tumor fragments were sutured to the outer surface of the GB in syngeneic mice, which developed orthotopic tumors that resembled human GB cancer in histological and transcriptional features. This model revealed the infiltration of similar subsets of immune cells in both subcutaneous and orthotopic tumors, confirming the appropriate immune environment during carcinogenesis. In addition, we accurately validated the in vivo efficacy of gemcitabine, a common therapeutic agent for GB cancer, in large cohorts. Taken together, this model may serve as a promising avatar of patients with GB cancer in drug discovery and precision medicine.

A dermoid cyst misdiagnosed as a lipoma due to atypical magnetic resonance images: a case report.

BACKGROUND: Dermoid cysts are well-known lesions that manifest as subcutaneous tumors around the lateral sides of the eyebrows in young patients. Computed tomography or magnetic resonance imaging (MRI) is often performed to confirm the diagnosis. On the other hand, a lipoma is usually a circular lesion, which is sometimes observed in the upper part of the face. The signals of both T1-weighted and T2-weighted images of MRI of a lipoma are, in general, relatively highly homogenous, and the signals decrease in fat-suppressed images. Therefore, differential diagnosis between a dermoid cyst and a lipoma is usually made with MRI, especially based on fat-suppressed images. Here, we present a case of misdiagnosis of a dermoid cyst as a lipoma because of atypical magnetic resonance images. CASE PRESENTATION: We report a case of a 24-year-old Japanese woman with a dermoid cyst around the lateral edge of the eyebrow. The cyst had been gradually increasing in size for the past 2 years. On MRI, it showed high internal signals on T1- and T2-weighted images. However, the signal intensity decreased homogeneously in the fat-suppressed T2-weighted images. The observed tumor had a yellowish appearance under the endoscope. On the basis of these findings, the lesion was considered a lipoma until it ruptured intraoperatively. The pathological diagnosis confirmed it to be a dermoid cyst. CONCLUSION: Some dermoid cysts contain lipid-rich liquid, and these may be misdiagnosed as lipomas by MRI. When a tumor is located at a common site for a dermoid cyst, the MRI images should be validated carefully if it appears like a lipoma, and the differential diagnosis should be considered carefully.

A Novel Approach to Subcutaneous Collecting Lymph Ducts Using a Small Diameter Wire in Animal Experiments and Clinical Trials.

Background: While performing microsurgery, including lymphaticovenous anastomosis (LVA) for chronic limb lymphedema, it is a common procedure to identify the subcutaneous collecting lymph ducts with near-infrared fluorescence lymphangiography (NIR) using indocyanine green. However, due to limitations such as minimum observable depth, only a few lymphatic ducts can be identified with this procedure. Hence, we developed a new smaller-diameter "lymphatic wire" (LW) that could be inserted directly into lymphatic collecting ducts of the limbs, enabling accurate identification and localization. Methods and Results: First, used the LW on the hind limbs of 6 swine, and 36 porcine lymphatic collecting ducts were identified, the outer diameter of which varied from 0.3-0.7 mm (mean 0.41 ± 0.11 mm). We could insert the LW after creating a side opening in 30 of these ducts. We encountered no difficulties during the procedure. In the pathological examination, adverse events such as valve dysfunction and perforation were not identified. Based on the results, a clinical evaluation of the LW was performed in two patients with lower extremity lymphedema, and the LW helped us identify lymphatic ducts in the subcutaneous layer, even at the sites where the NIR had proved ineffective. Conclusion: Based on our results, we suggest that the procedure for identifying lymphatic vessels using the newly developed LW is a useful technique that can be utilized before performing a LVA for lymphedema. However, further clinical study is required to develop this device and technique, for wider clinical application in the future.

Le fort II distraction osteogenesis with a hybrid system for an Apert syndrome patient: A case report.

BACKGROUND: Le Fort II advancement is considered for normalizing the facial appearance in Apert syndrome. When these procedures are performed during growth, overcorrection of midface advancement is required. We developed a system that can control the distance and vector of movement for the central midface to create more normal facial proportions. This case report shows Le Fort II distraction osteogenesis with this hybrid system for an Apert syndrome patient. CASE: The patient was a girl with Apert syndrome with midfacial-nose hypoplasia and skeletal class III malocclusion. She was healthy without respiratory problems and had no learning disabilities. She underwent our Le Fort II distraction osteogenesis with the hybrid system at 10 years and 6 months of age. Her midface was elongated 22 mm at point Or forward and moved 5° downward to the Frankfort horizontal plane compared to the standard position of average Japanese adult women on the cephalogram. Examining the facial image, the midfacial depression was improved 4 years after the operation. DISCUSSION: Overcorrection of midface advancement is required for patients to reduce the number of procedures during growth. The system that we developed could control the distance and vector of movement steadily when the central midface was overcorrected to try to create normal adult facial proportions.

Intraoral Approach for a Congenital Teratoma in the Orbit Extending into the Pterygopalatine Fossa.

This article reports an intraoral approach for a congenital mature teratoma in the orbit, extending into the pterygopalatine fossa and the zygoma remodeling due to biological and functional matrix theory during the neonatal period. The operation took place on the 56th postnatal day due to gradual proptosis of the left eye from the socket. The entire orbital tumor could be surgically removed via the inferior orbital fissure, which had been expanded by the pressure of the tumor. This case suggests that orbital tumors expanding into pterygopalatine fossa can be removed by the intraoral approach because the bone of infants is so soft. The eyeball of the patient was temporarily depressed after the operation, but that was improved by tissue remodeling with time. Therefore, it is recommended that reconstructive surgery for an infant should be performed at the age of 12 months or older.

Development of a Method for Scaffold-Free Elastic Cartilage Creation.

Microtia is a congenital aplasia of the auricular cartilage. Conventionally, autologous costal cartilage grafts are collected and shaped for transplantation. However, in this method, excessive invasion occurs due to limitations in the costal cartilage collection. Due to deformation over time after transplantation of the shaped graft, problems with long-term morphological maintenance exist. Additionally, the lack of elasticity with costal cartilage grafts is worth mentioning, as costal cartilage is a type of hyaline cartilage. Medical plastic materials have been transplanted as alternatives to costal cartilage, but transplant rejection and deformation over time are inevitable. It is imperative to create tissues for transplantation using cells of biological origin. Hence, cartilage tissues were developed using a biodegradable scaffold material. However, such materials suffer from transplant rejection and biodegradation, causing the transplanted cartilage tissue to deform due to a lack of elasticity. To address this problem, we established a method for creating elastic cartilage tissue for transplantation with autologous cells without using scaffold materials. Chondrocyte progenitor cells were collected from perichondrial tissue of the ear cartilage. By using a multilayer culture and a three-dimensional rotating suspension culture vessel system, we succeeded in creating scaffold-free elastic cartilage from cartilage progenitor cells.

HAMAMATSU-ICG study: Protocol for a phase III, multicentre, single-arm study to assess the usefulness of indocyanine green fluorescent lymphography in assessing secondary lymphoedema.

INTRODUCTION: Secondary lymphoedema of the extremities is an important quality-of-life issue for patients who were treated for their malignancies. Indocyanine green (ICG) fluorescent lymphography may be helpful for assessing lymphoedema and for planning lymphaticovenular anastomosis (LVA). The objective of the present clinical trial is to confirm whether or not ICG fluorescent lymphography using the near-infrared monitoring camera is useful for assessing the indication for LVA, for the identification of the lymphatic vessels before the conduct of LVA, and for the confirmation of the patency of the anastomosis site during surgery. METHODS AND ANALYSIS: This trial is a phase III, multicentre, single-arm, open-label clinical trial to assess the efficacy and safety of ICG fluorescent lymphography when assessing and treating lymphoedema of patients with secondary lymphoedema who are under consideration for LVA. The primary endpoint is the identification rate of the lymphatic vessels at the incision site based on ICG fluorescent lymphograms obtained before surgery. The secondary endpoints are 1) the sensitivity and specificity of dermal back flow determined by ICG fluorescent lymphography as compared with 99mTc lymphoscintigraphy-one of the standard diagnostic methods and 2) the usefulness of ICG fluorescent lymphography when confirming the patency of the anastomosis site after LVA. ETHICS AND DISSEMINATION: The protocol for the study was approved by the Institutional Review Board of each institution. The trial was filed for and registered at the Pharmaceuticals and Medical Devices Agency in Japan. The trial is currently on-going and is scheduled to end in June 2020. TRIAL REGISTRATION NUMBER: jRCT2031190064; Pre-results.

Low-density-lipoprotein apheresis-mediated endothelial activation therapy to severe-peripheral artery disease study: Rationale and study design.

A novel approach is required for standard therapy-resistant peripheral arterial disease (PAD). This is a single-center, single-arm, interventional study (LDL Apheresis-Mediated Endothelial Activation Therapy to Severe-Peripheral Artery Disease study), which aims to evaluate the efficacy and safety of lipoprotein apheresis (LA) with a dextran sulfate cellulose column in PAD with controlled serum cholesterol levels. The study participants have standard therapy-resistant PAD with controlled serum cholesterol levels. A total of 35 patients undergo 10 sessions of LA therapy. The ankle-brachial index and vascular quality of life questionnaire are assessed before and after the treatment period as primary outcomes. Registration of patients began in November 2015 and is planned to be concluded in October 2020.

Cardiac overload resolved by resection of a large plexiform neurofibroma on both the buttocks and upper posterior thighs in a patient with neurofibromatosis type I: a case report.

BACKGROUND: A large plexiform neurofibroma in patients with neurofibromatosis type I can be life threatening due to possible massive bleeding within the lesion. Although the literature includes many reports that describe the plexiform neurofibroma size and weight or strategies for their surgical treatment, few have discussed their possible physical or mental benefits, such as reducing cardiac stress. In addition, resection of these large tumors can result in impaired wound healing, partly due to massive blood loss during surgery. CASE PRESENTATION: A 24-year-old man was diagnosed with neurofibromatosis type I and burdened with a large plexiform neurofibroma on the buttocks and upper posterior thighs. The patient was 159 cm in height and 70.0 kg in weight at the first visit. Cardiac overload was indicated by an echocardiography before surgery. His cardiac output was 5.2 L/min with mild tricuspid regurgitation. After embolism of the arteries feeding the tumor, the patient underwent surgery to remove the neurofibroma, followed by skin grafting. Follow-up echocardiography, performed 6 months after the final surgery, indicated a decreased cardiac output (3.6 L/min) with improvement of tricuspid regurgitation. Because the blood loss during the first surgery was over 3.8 L, malnutrition with albuminemia was induced and half of the skin graft did not attach. Nutritional support to improve the albuminemia produced better results following a second surgery to repair the skin wound. CONCLUSION: Cardiac overload may be latent in patients with neurofibromatosis type I with large plexiform neurofibromas. As in pregnancy, the body may compensate for this burden. In these patients, one stage total excision may improve quality of life and reduce cardiac overload. In addition, nutritional support is likely needed following a major surgery that results in either an extensive skin wound or excessive blood loss during treatment.

Extended Maxillotomy for Nasopharynx Access in Infantile Immature Teratoma.

Infantile immature teratoma located in the nasopharynx is a rare congenital tumor that is not easily removed. Three surgeries and chemotherapy for recurrence of the tumor have been performed since a male infant with a nasopharyngeal mass was born at a gestational age of 35 weeks. Extended maxillotomy combining Le Fort I osteotomy with midline palatal split was performed at 2 years and 6 months of age. Residual tumor left in the intracranial region had not increased as of 4 years of age. Careful follow-up is needed until the patient reaches adulthood.

Pathological changes in the lymphatic system of patients with secondary upper limb lymphoedema.

Secondary upper limb lymphoedema is usually caused by lymphatic system dysfunction. Diagnosis is primarily based on clinical features. However, there are no distinct diagnostic criteria for lymphoedema. Although conventional lymphoscintigraphy is a useful technique to diagnose the severity of lymphoedema, the resultant data are two-dimensional. In this study, we examined the pathology of lymphoedema using single photon emission computed tomography-computed tomography lymphoscintigraphy (SPECT-CT LSG), a new technique that provides 3-dimensional information on lymph flow. We observed lymph flow pathways in the subcutaneous and muscle layers of the upper limbs. A significant positive correlation was found between the dermal back flow (DBF) type and the visualization of lymph nodes around the clavicle (p = 0.000266), the type of lymph flow pathways and the visualization of lymph nodes around the clavicle (p = 0.00963), and the DBF type and the lymph flow pathway (p = 0.00766). As the severity of lymphoedema increased, the DBF appeared more distally in the upper limb and the flow into the lymph nodes around the clavicle decreased, whereas the lymph flow pathways in the muscle layer became dominant. These findings demonstrate the features of lymphoedema pathology and the functional anatomy and physiology of the lymphatic system without the need for cadaver dissection.

Overcorrected Midface Advancement to Improve Airway Problems in Severe Pfeiffer Syndrome Types II and III.

INTRODUCTION: Treatment of patients with severe Pfeiffer syndrome types II and III is difficult. The purpose of this article is to present our method of overcorrecting midface advancement to improve airway problems in such patients. MATERIALS AND METHODS: One boy and two girls with types II and III Pfeiffer syndrome and who underwent Le Fort III midface advancement using our previously described corrected cephalometric analysis and distraction system were included in the study. RESULTS: The authors overcorrected by advancing the midface to make it look as similar as possible to an adult face. While the overcorrected midface advancement widened the upper airway spaces in the 3 patients, the tracheostomy that had already been placed during infancy could not be closed, probably because of an underlying tracheal abnormality or tracheomalacia. DISCUSSION: Overcorrected midface advancement cannot enable tracheostomy closure, probably because of severe tracheal anomalies, such as tracheomalacia, below the tracheostomy. However, with the possibility of gradual improvement of the tracheomalacia with age, closure of the tracheostomy can eventually be expected. Therefore, efforts to close a tracheostomy should be pursued even if the probability of its removal is low. CONCLUSION: Overcorrected midface advancement did not enable tracheostomy closure, probably because of severe tracheal anomalies such as tracheomalacia. However, the severe exophthalmos and angle III malocclusion were improved, and with the possibility of gradual improvement of the tracheomalacia with age, closure of the tracheostomy can eventually be expected. Therefore, efforts to close a tracheostomy should be pursued even if the probability of its removal is low.