Fate of Costochondral Grafts for Micrognathia: 3D Analysis of Postoperative Length, Position, and Graft Take.

PURPOSE: Costochondral grafting is performed in the reconstruction of severe micrognathia. In this study, we report our experience with graft take, as well as unique findings that have not been focused on in the past literature such as postoperative position and changes in the shape of the graft and thinning of the skull base in contact with the graft. METHODS: Between 2002 and 2019, costochondral grafting was performed on 15 sides of 10 patients with micrognathia. Graft take and other detailed findings such as growth, position, and changes in the shape of the grafts and skull base in contact with the grafts were evaluated using computed tomography images. RESULTS: Graft take was obtained in all cases, and subsequent distraction could be performed in 8 of 10 graft sites as planned. Six of 15 grafts were displaced anterior to the preoperative planned position. However, there were no cases with complications due to deviation such as trismus. Concave deformity of the skull base in contact with the graft was observed in 9 graft sites. The thinning and bending deformation were observed in 2 grafts. CONCLUSIONS: Costochondral grafting was considered a reasonable method for first-line treatment of severe micrognathia. Several unique findings that have not been focused on in the past literature were observed. These should be kept in mind when performing costochondral grafting. Several modifications to the present protocol are suggested to improve the quality of graft take.

Clinical Features of Bilambdoid and Sagittal Synostosis (BLSS): A Retrospective Multicenter Study in Japan.

ABSTRACT: Bilambdoid and sagittal synostosis (BLSS), a rare form of multisutural craniosynostosis, is sometimes known as the Mercedes-Benz syndrome due to the appearance of the fused sagittal and bilateral lambdoid sutures. Although previous studies have described some of its clinical features, the pathology of this disease is not yet fully understood. Moreover, it has been pointed out that BLSS is more common among individuals of Hispanic ethnicity, but its incidence in Asia remains unclear. In the present study, BLSS cases in Japan were analyzed to determine the characteristics of the condition in Japan. Three hospitals in Tokyo participated in the present study. Patients with BLSS who underwent cranial remodeling were included. Data on patient demographics, clinical symptoms, status of the cranial sutures, morphological subclassification, surgical procedures, developmental status, and genetic mutations were analyzed. In total, 22 patients met the enrollment criteria and were included, indicating a higher incidence of BLSS in Japan than in other nations reported in previous studies. In terms of morphological subclassification, there were 15 brachycephalic, 4 dolichocephalic, and 3 normocephalic. For the initial cranial procedure, 7 patients underwent a single-stage cranioplasty, 13 underwent a posterior distraction, and 2 underwent lateral expansion. Patients with a normocephalic cranial morphology tended to undergo surgery at an older age than patients with the other two types. Appropriate timing for surgery is important for healthy development; hence, surgeons should remember that BLSS can lead to "balanced dysmorphism" that may have led to a delay in diagnosis due to its normal-looking morphology.

Ecthyma gangrenosum in a 3-year-old boy post-heart transplantation.

Ecthyma gangrenosum (EG) is a serious bacterial infection in immunocompromised patients. EG in transplant recipients is rarely reported and may go unrecognized, which may delay initiation of appropriate treatment. We report a case of EG in a pediatric heart transplant recipient who was treated successfully with antibiotics and surgical debridement.

The Incidence of Sleep Disordered Breathing One Week After Primary Palatoplasty: Evaluation With Overnight Pulse Oximetry.

BACKGROUND: Sleep disordered breathing (SDB) is defined as a series of disorders including snoring, obstructive sleep apnea, and hypopnea. Few studies investigated the incidence of SDB following primary palatoplasty with objective testing. The aims of this study were to elucidate the prevalence and degree of SDB approximately 1 week following primary palatoplasty with objective testing and to clarify the risk factors. METHOD: A retrospective review was performed on children who underwent primary palatoplasty between April 2013 and July 2017 at National Center for Child Health and Development, Tokyo, Japan. As a national center, the authors accept many syndromic patients. The authors keep all patients after palatoplasty intubated and observe them overnight in intensive care unit to reduce the risks of respiratory events. Patients were evaluated with overnight pulse oximetry on 5 to 7 days postoperatively. RESULTS: Forty-four patients were included, and 30% of the patients were associated with congenital anomaly. Thirteen patients (30%) were diagnosed with SDB. None of the patients required additional treatment after the evaluation. Laryngomalacia and postoperative oxygen requirement significantly correlated with postoperative SDB. CONCLUSION: Approximately one-third of the patients may be at the risk of SDB 1 week after primary palatoplasty. Patients with history of laryngomalacia or those who required oxygen support for prolonged time after primary palatoplasty should be cared for significantly high risk of postoperative SDB.

Analysis of Cranial Morphology of Healthy Infants Using Homologous Modeling.

OBJECTIVE: Data on cranial morphology of healthy individuals can be used as the guide in the treatment of cranial deformity. There are many reports analyzing the cranial morphology of healthy children in the past. But most of them focus on 2-dimensional values, and there are only a few reports, which analyzed the cranial morphology of Japanese healthy infants. We report a novel method that enables the comprehensive analysis of cranial morphology of Japanese healthy infants in 3D. METHODS: Craniofacial CT data of 20 healthy infants (9 males, 11 females) ranging in age from 1 to 11 months were collected. Based on the CT data, we created 20 homologous models of cranium using software specifically designed to support homologous modeling. We averaged vertex coordinates of the homologous models to create average model. We further performed principal component analysis, and created virtual models based on each principal component. The contribution rate was calculated, and the features described by each principal component were interpreted. RESULTS: We created the average cranial model of Japanese healthy infants. Seven principal components (cumulative contribution rate: 89.218%) were interpreted as to which part of the cranial shape each component was related to. The elements were extracted that may characterize the cranial morphology of some of the clinical conditions such as dolico/brachycephaly and deformational plagiocephaly. Some of these elements have not been mentioned in the past literature. CONCLUSION: Homologous modeling was considered to be valid and strong tool for comprehensive analysis of cranial morphology.

Reconstruction of the Urethral Sphincter with Dynamic Graciloplasty in a Male Rabbit Model.

INTRODUCTION: The use of artificial urinary sphincters can improve urinary incontinence after radical prostatectomies; however, complications can arise. We hypothesized that dynamic graciloplasty improves urethral sphincter reconstruction. MATERIALS AND METHODS: Models of urethral sphincter muscle reconstruction were developed in 5 adult rabbits by wrapping the gracilis muscle flap around the urethra. Intra-urethral pressure was measured in each of the models before reconstruction (control), after reconstruction, and after electrical stimulation of the flap in reconstructed models (stimulated models). RESULTS: The mean maximum urethral closure pressure was significantly greater in the reconstruction model (69.7 (66.5-115.8) mm Hg) than in the control model (39.2 (33.7-49.6) mm Hg). The mean integral of the urethral pressure and urethral length was also significantly greater in the reconstruction model than in the control model. Furthermore, sphincter tightening was enhanced by the electrical stimulation of the flap. CONCLUSIONS: Our results support our hypothesis that the functional reconstruction of urethral sphincters using muscle flaps is promising for the treatment of urinary incontinence.

Anatomic basis of anorectal reconstruction by dynamic graciloplasty with pudendal nerve anastomosis.

BACKGROUND: Dynamic graciloplasty has been proposed for anal reconstruction, but this method has 2 major drawbacks. First, an electrical device is required for control of the gracilis. The anastomosis with the pudendal nerve will provide more physiological control. Second, the limitation in the mobility of the muscle flap results in wrapping the anal canal with the muscle's distal portion, which is tendonlike and inelastic. Enhancing the mobility of the muscle flap will enable wrapping with the proximal, muscle-like, and extensible portion, possibly providing better sphincteric function. However, the basis for such an operative method is lacking. OBJECTIVE: The aim of this study is to provide the basis for the refined method of anal sphincter reconstruction by dynamic graciloplasty with pudendal nerve anastomosis and to verify the feasibility of lengthening the nerve to the gracilis muscle flap by dissecting into the muscle belly, detaching the gracilis muscle from its origin, and enhancing the mobility of the muscle flap. STUDY DESIGN: This is a retrospective, descriptive study. METHODS: The results from the anatomical study on 9 cadavers are reported. RESULTS: Tension-free anastomosis of the pudendal nerve and nerve to the gracilis was successfully performed in all the 9 cases: in 2 cases, by lengthening the nerve. The detachment of the muscle origin improved the mobility of the muscle flap, and the more proximal portion could be used for wrapping the anal canal, as confirmed in 4 cases. LIMITATIONS: The limited number of cases was a shortcoming of this study. CONCLUSIONS: By lengthening the nerve to the muscle, the gracilis can be used for anal sphincter reconstruction with pudendal nerve anastomosis, negating the need for an electrical device. By detaching the origin of the gracilis muscle, its proximal portion can be used to wrap the anal canal, possibly enabling a longer functional canal with stronger constricting force and better vascularity. These modifications to past methods may improve fecal continence after the operation.

Evaluation of maxillary sinus volume in cleft alveolus patients using 3-dimensional computed tomography.

OBJECTIVE: The aim of the study was to evaluate the volume of the maxillary sinus in patients with cleft alveolus. STUDY DESIGN: This is a retrospective, descriptive study. PATIENTS AND METHODS: The 3-dimensional computed tomographic data of 218 maxillary sinuses of 109 patients with cleft alveolus were compared with those of 100 sinuses of 50 healthy individuals. RESULTS: No significant difference in the maxillary sinus volume was found between the patients with cleft alveolus and the noncleft individuals. In the patients with cleft palate and alveolus, the maxillary sinus volume was significantly larger on the right side, but no significant difference was found between the cleft and noncleft sides. CONCLUSIONS: The volume of the maxillary sinus in the patients with cleft alveolus is not different from that of the noncleft individuals. The information about the maxillary sinus is clinically important in executing such operations as endoscopic sinus surgery.

Necrotizing fasciitis following rapidly deteriorating neonatal omphalitis with good initial presentation.

Neonatal omphalitis is a postpartum infection of periumbilical superficial soft tissues that usually has a good prognosis in developed countries. In rare cases, it could progress to periumbilical necrotizing fasciitis (NF), which is an infection of the deep soft tissues, including muscle fascia, and has a high mortality rate. However, the signs and timing of developing NF secondary to omphalitis are unclear. We encountered a neonatal case of NF following omphalitis. In the initial days of the clinical course, general symptoms and condition of the patient were good, and abdominal physical findings were mild; however, the patient rapidly developed NF. The patient was successfully treated by emergent surgical debridement, broad-spectrum antibiotics, and intensive care. To determine the area of blood perfusion, we intravenously injected indocyanine green by intraoperative angiography, and then extensively removed necrotic and hypoperfused tissues. In neonatal omphalitis, the deterioration can suddenly occur despite good initial conditions; intensive monitoring should be required during the first few days of the clinical course.

Modified Urethral Graciloplasty Cross-Innervated by the Pudendal Nerve for Postprostatectomy Urinary Incontinence: Cadaveric Simulation Surgery and a Clinical Case Report.

An artificial sphincter implanted in the bulbous urethra to treat severe postprostatectomy urinary incontinence is effective, but embedding-associated complications can occur. We assessed the feasibility, efficacy, and safety of urethral graciloplasty cross-innervated by the pudendal nerve. A simulation surgery on three male fresh cadavers was performed. Both ends of the gracilis muscle were isolated only on its vascular pedicle with proximal end of the obturator nerve severed and transferred to the perineum. We examined whether the gracilis muscle could be wrapped around the bulbous urethra and whether the obturator nerve was long enough to suture with the pudendal nerve. In addition, surgery was performed on a 71-year-old male patient with severe urinary incontinence. The postoperative 12-month outcomes were assessed using a 24-hour pad test and urodynamic study. In all cadaveric simulations, the gracilis muscles could be wrapped around the bulbous urethra in a γ-loop configuration. The length of the obturator nerve was sufficient for neurorrhaphy with the pudendal nerve. In the clinical case, the postoperative course was uneventful. The mean maximum urethral closure pressure and functional profile length increased from 40.7 to 70 cm H 2 O and from 40.1 to 45.3 mm, respectively. Although urinary incontinence was not completely cured, the patient was able to maintain urinary continence at night. Urethral graciloplasty cross-innervated by the pudendal nerve is effective in raising the urethral pressure and reducing urinary incontinence.

Evaluation of the Molding Helmet Therapy for Japanese Infants with Deformational Plagiocephaly.

INTRODUCTION: Deformational plagiocephaly (DP) is cranial flattening on one side of the back of the skull produced by an extrinsic force on the intrinsically normal skull. When the flattening is symmetrical, the deformity is called deformational brachycephaly (DB). In the US, its prevalence has increased since the "Back to Sleep" campaign by the American Association of Pediatrics. Helmet therapy is reported to be effective in improving head deformity by multiple studies, but there are few evidences from Japan. The purpose of this study is to investigate the safety and efficacy of helmet therapy for DP, and the feasibility of introducing this treatment to the clinical setting in Japan. METHODS: This was a single-arm, retrospective, nonrandomized study. Data were collected on infants who visited the "Clinic for Baby's Head Shape" in the National Center for Child Health and Development, Tokyo, Japan, between 2011 and 2014. Improvements in Argenta classification, cranial asymmetry (CA), and cranial vault asymmetry index (CVAI) were evaluated. The relationships between CA and influencing factors were evaluated using a linear mixed-effects model. RESULTS: Three hundred eighty-seven infants (273 boys and 114 girls; average age, 4.7 months) visited the clinic during the period, and 159 patients who completed the helmet therapy were analyzed. There were statistically significant improvements in Argenta classification, CA, and CVAI. Almost all of the parents reported increased sweating and mild skin irritation, but no adverse events necessitated the cessation of helmet therapy, except for one patient with increased sweating. CONCLUSIONS: Helmet therapy is safe and effective in treating DP and is feasible to introduce to the clinical setting in Japan. Through the distribution of knowledge regarding the etiology and treatment of head deformity, earlier detection and an evidence-based approach to head deformity are expected in the future.

Protocol for a multicentre, prospective, cohort study to investigate patient satisfaction and quality of life after immediate breast reconstruction in Japan: the SAQLA study.

INTRODUCTION: The aim of breast reconstruction (BR) is to improve patients' health-related quality of life (HRQOL). Therefore, measuring patient-reported outcomes (PROs) would clarify the value and impact of BR on a patient's life and thus would provide evidence-based information to help decision-making. The Satisfaction and Quality of Life After Immediate Breast Reconstruction study aimed to investigate satisfaction and HRQOL in Japanese patients with breast cancer who undergo immediate breast reconstruction (IBR). METHODS AND ANALYSIS: This ongoing prospective, observational multicentre study will assess 406 patients who had unilateral breast cancer and underwent mastectomy and IBR, and were recruited from April 2018 to July 2019. All participants were recruited from seven hospitals: Okayama University Hospital, Iwate Medical University Hospital, The Cancer Institute Hospital of Japanese Foundation for Cancer Research, Showa University Hospital, University of Tsukuba Hospital, Osaka University Hospital and Yokohama City University Medical Center. The patients will be followed up for 36 months postoperatively. The primary endpoint of this study will be the time-dependent changes in BREAST-Q satisfaction with breast subscale scores for 12 months after reconstructive surgery, which will be collected via an electronic PRO system. ETHICS AND DISSEMINATION: This study will be performed in accordance with the Ethical Guidelines for Medical and Health Research Involving Human Subjects published by Japan's Ministry of Education, Science and Technology and the Ministry of Health, Labour and Welfare, the modified Act on the Protection of Personal Information and the Declaration of Helsinki. This study protocol was approved by the institutional ethics committee at the Okayama University Graduate School of Medicine, Dentistry, on 2 February 2018 (1801-039) and all other participating sites. The findings of this trial will be submitted to an international peer-reviewed journal. TRIAL REGISTRATION NUMBER: UMIN000032177.

Average Models and 3-dimensional Growth Patterns of the Healthy Infant Cranium.

Treatment of cranial deformity is often performed during infancy in cases such as craniosynostosis and deformational plagiocephaly. To acquire morphologic standards for the treatment goals of these conditions, we created cranial average models and elucidated the growth patterns of the cranium of healthy infants in 3-dimension (3D) using homologous modeling. METHODS: Homologous modeling is a technique that enables mathematical analysis of different 3D objects by converting the objects into homologous models that share the same number of vertices with the same spatial relationships. Craniofacial computed tomographic data of 120 healthy infants ranging in age from 1 to 17 months were collected. Based on the computed tomographic data, we created 120 homologous models. Six average 3D models (20 individuals each for 6 different age groups) were created by averaging the vertices of the models. Three-dimensional growth patterns of the cranium were clarified by comparing the 6 average models. RESULTS: We successfully created 6 average models and visualized the growth patterns of the cranium. From 1-month-old to 5-month-old infants, the entire cranium except for the occipital region grows, and the cranium tended to be brachycephalic (cephalic index at 4-5 months: 87.1-97.3), but the growth was thereafter localized to specific areas. CONCLUSIONS: Three-dimensional growth patterns of the cranium of healthy infants were clarified. These findings will support the understanding and treatment of the conditions that cause cranial deformity. To our knowledge, this is the first report to visualize the growth patterns of the entire cranium of healthy infants in 3D.

A new method to quantify subtle morphological deformities in nasal profile curvatures and its application for analysis of unilateral cleft lip noses.

OBJECTIVE: The authors conducted the present study to elucidate what elements characterize the nasal profiles of patients with unilateral cleft lips (CLs). MATERIALS AND METHODS: A total of 40 Japanese unilateral CL patients were studied. For each patient, the nasal profile curve was traced on three-dimensional computer tomography image. Then four points were marked on the contour. The points were NAS (Nasion), MAP (the Most Anterior Point on the nasal profile curve), GPRN (the Genuine Pronasale: the point on the nasal curve at which the curve protrudes most), and SBN (Subnasale: the point at the columellar base). Using specially designed software, the distances between these marking points were measured along the nasal profile curve. RESULTS: In CL patients, the distance between the MAP and GPRN is longer, and the GPRN is located more inferiorly than in non-cleft persons. CONCLUSION: The nasal tip tends to become round and to droop in unilateral CL patients. In order to avoid this deformity pattern, the nasal tip should be reshaped to present a sharper curvature and corrected superiorly.

Analysis of the orbital floor morphology.

INTRODUCTION: For the repair of large orbital floor defects due to blow-out fractures or those involved by tumours, it is necessary to understand the detailed morphology. The purpose of the present study was to elucidate how age and gender affect its three-dimensional morphology. METHODS: The 3-D computer tomography data of 305 orbits of 182 patients were included in the study. Using the CT data, the orbital floor angle (the angle between the orbital floor and the horizontal plane) and the location of the most inferior point of the orbital floor were measured. Patients were classified into subgroups according to their gender and age. Data were compared among the subgroups. RESULTS: The orbital floor angle was greater in males than in females, and in children than in adults. The location of the lowest point of the orbital floor moves postero-inferiorly with increasing age. CONCLUSION: The present study demonstrated that gender and age affects morphology of the orbital floor. These findings should be useful for reconstruction of the orbital floor.

Appropriate diameter for screws to fix the maxilla following Le Fort I osteotomy: an investigation utilizing finite element analysis.

BACKGROUND: After Le Fort I osteotomy, there is sometimes a secondary deformity (relapse), with the lower segment deviating from the intraoperatively fixed position. It is hyopothesized that the structural stability of the reconstructed maxilla is affected by the diameter of the fixation screws. The present study aims to elucidate the relationship between the diameters of the screws and the structural stability of the maxilla after Le Fort I osteotomy. METHODS: 3D models were produced on a workstation from 20 dry skulls and a Le Fort I operation was simulated on them. The upper and lower segments of the divided maxilla in each of the 20 models were connected using four plates and 16 screws. Five different diameters of the fixation screws were tested. Thus altogether 100 models were produced. A 180N load was applied to the molar region for each model. Using finite element analysis, the resultant stresses and deviations of the lower segments were calculated. Finally, referring to these values, the relationships between screw diameters and stability of the lower segment were evaluated. RESULT: The stability of the lower segment was greatest when the diameter of the fixation screws was equal to the thickness of the bone at each fixation site. CONCLUSION: In performing Le Fort I osteotomy, it is recommended that bone thickness is measured at each fixation site in advance, and the diameter of the fixation screws matched accordingly; thereby optimum stability of the reconstructed maxilla can be anticipated.

Iodine 123-metaiodobenzylguanidine imaging reflect generalized sympathetic activation in patients with left ventricular dysfunction.

BACKGROUND: Iodine 123-metaiodobenzylguanidine (MIBG) imaging has been used to assess cardiac sympathetic nerve abnormalities. To determine the role of MIBG imaging as a measure of generalized sympathetic nerve activity, MIBG imaging was evaluated with muscle sympathetic nerve activity (MSNA) and plasma norepinephrine (noradrenaline) level in patients with old myocardial infarction. METHODS: Myocardial MIBG scintigraphy, MSNA and plasma norepinephrine measurement were performed within 3 days in 35 patients with old myocardial infarction. Patients were divided into three groups according to their ejection fraction (EF); preserved (EF > or = 50%, 12 patients), intermediate (35% < EF < 50%, 13 patients), or depressed (EF < or = 35%, 10 patients). The heart to mediastinum (H/M) ratio was obtained 4 h after tracer injection from the chest anterior view image. MIBG washout rate was calculated from the early and delayed images. MSNA was recorded by microneurography. RESULTS: Plasma norepinephrine level had weak negative correlations with early H/M ratio (r = 0.37, P<0.05) and delayed H/M ratio (r = 0.33, P<0.05), and a positive correlation with MIBG washout rate (r = 0.54, P<0.01). MSNA had weak negative correlations with early H/M ratio (r = 0.51, P<0.05) and delayed H/M ratio (r = 0.52, P<0.05). However, a strong correlation was found between MSNA and MIBG washout rate (r = 0.88, P<0.001). Despite no significant differences in plasma norepinephrine level and H/M ratio, patients with intermediate and depressed EF had higher MIBG washout rate and MSNA compared with those with preserved EF. CONCLUSIONS: Increased in cardiac sympathetic nerve activity was associated with augmented sympathetic outflow of the skeletal muscle and hence, MIBG washout rate allow the assessment of general sympathetic nerve activity.

P-selectin and platelet-derived microparticles associated with monocyte activation markers in patients with pulmonary embolism.

Platelet activation markers (platelet-derived microparticles and P-selectin on activated platelets), chemokines (monocyte chemotactic peptide and regulated on activation normally T-cell expressed and secreted), and soluble markers (sP-selectin, sE-selectin, sVCAM-1, and sCD14) were measured and compared in patients with pulmonary embolism (PE). These substances are thought to participate in the pathogenesis of PE. Levels of all of the platelet activation markers, chemokines, and soluble markers were higher in the patients with PE than in normal controls. Levels of platelet activation markers were also significantly increased postoperatively after total knee arthroplasty. Anti-platelet therapy significantly inhibited the elevation of platelet activation markers after total knee arthroplasty. These findings suggest that antiplatelet therapy may be useful for PE-related interaction of platelets, leukocytes, and endothelial cells.

QOL after head and neck reconstruction: evaluation of Japanese patients using SF-36 and GOHAI.

OBJECTIVE: To evaluate the health-related quality of life (HRQOL) of Japanese patients with head and neck cancer after treatment. STUDY DESIGN: Cross-sectional, descriptive study. PATIENTS AND METHODS: Twenty-nine patients who underwent resection and reconstruction of the head and neck cancer between September 2001 and January 2008 at the National Hospital Organization Tokyo Medical Center completed the Short Form 36 (generic QOL measure) and the General Oral Health Assessment Index (oral-specific QOL measure). RESULTS: The generic QOL of the patients was relatively maintained while oral-specific QOL was impaired compared to the Japanese norms. The patients with musculo-cutaneous flaps and 1y or longer after operation reported significantly lower QOL. CONCLUSION: This is a unique study on Japanese patients with relatively longer time after operation. Further evaluation with increased number of cases and disease-specific QOL scale is required to better understand the QOL of the patients.

Refined simple line closure for macrostomia repair: designing a mucosal triangular flap on the commissure region.

Congenital macrostomia is a relatively rare deformity. A number of different methods for its correction have been reported in the past. Here, we report our refined method of correcting macrostomia. Our method is characterized by creation of a small triangular mucosal flap on the cleft region of the lower red lip where the tissue appears identical to the normal commissure. Adequate advancement of this flap into the mouth along with proper reconstruction of muscle continuity and simple line closure of the skin enable correction of macrostomia with a natural-looking commissure in a symmetric position with an acceptable scar.