Management of MACROGLOSSIA: Case Series and Suggested Algorithm.

PURPOSE: Idiopathic macroglossia is a rare entity of true tongue enlargement without an underlying etiology. There are only a few case reports on the diagnosis and management of idiopathic macroglossia. This study's purpose was to present a series of patients with idiopathic macroglossia and suggest a treatment algorithm. METHODS: This was a retrospective case series of a cohort of patients with macroglossia who were treated by the Oral and Maxillofacial Surgery service at the University of Texas Health Science Center at Houston (UTHealth)and Emory University. The patient's medical comorbidities, history of present illness, clinical presentation, radiographic findings, and disease management were studied. The outcome variables include normalization of the tongue size, dependence on parenteral nutrition, and tolerating tracheostomy decannulation. RESULTS: Five patients with a mean age of 45 years were included in the study. All of the patients (n = 5, 100%) in our cohort developed macroglossia following prolonged oral intubation, with 3.5 weeks being the average length of intubation. All patients presented with difficulty feeding orally and breathing. The average tongue dimension was 12.20 x 6.25 cm. All tongue enlargements were located in the anterior 2/3 of the tongue, and all patients had displaced anterior dentition. In addition, 60% of the patients (n = 3) experienced altered tongue sensation (pain and/or decreased taste). These patients were surgically managed with tracheostomy and percutaneous endoscopic gastrostomy (PEG) tube placement followed by partial glossectomy (n = 5, 100%). We defined successful outcomes as 1) modifying the tongue to a functional, nonprotruding form, 2) tracheostomy decannulation and 3) PEG tube removal. Tracheostomy decannulation and PEG tube removal were achieved in 80% of the patients (n = 4). CONCLUSIONS: In this patient cohort, we were unable to identify the cause of the pathology based on existing clinical data. When the etiology is unclear or irreversible, management should involve tracheostomy and surgical feeding access for the initial stabilization, followed by modified glossectomy to improve form, function, and cosmesis thereby improving the overall quality of life.

COVID-Associated Avascular Necrosis of the Maxilla-A Rare, New Side Effect of COVID-19.

PURPOSE: The purpose of this article is to present an interesting, rare case of a patient who experienced avascular necrosis of the maxilla associated with COVID-19 infection. METHODS AND RESULTS: Our team retrospectively evaluated this patient's chart after completion of surgical management. The patient is a 72-year-old male who presented to the University of Texas Health Science Center at Houston for surgical management of his infarcted maxilla, which developed as a sequela of infection with COVID-19. A literature review was completed using PubMed. Twenty-five articles are reviewed and discussed. CONCLUSIONS: Infection with COVID-19 confers a hypercoagulable state in patients, leading to various complications in the head and neck region. In our case report, we present a patient who developed avascular necrosis of the maxilla secondary to infection with COVID-19. Thromboembolic prophylaxis is imperative in COVID-19 patients due to the high rate of potential systemic complications.

The Incidence of COVID-19 Patients in Oral and Maxillofacial Surgery.

PURPOSE: The SARS-CoV-2 global pandemic has resulted in widespread changes to healthcare practices across the United States. The purpose of this study is to examine the incidence of COVID-19 patients in the oral-maxillofacial surgery setting in order to help guide perioperative protocols during the pandemic. METHODS: In this retrospective cohort study, predictor variables (presence of preoperative symptoms on presentation, patient age, patient gender, patient race, hospital location, and presence of statewide stay-at-home orders) were examined with outcome variables (SARS-CoV-2 test results) over 10 months between March 2020 and December 2020 for patients undergoing surgical procedures in the operating room by the following Oral-Maxillofacial Surgery Departments: - Louisiana State University Health Sciences Center (Baton Rouge, LA) - University of Illinois at Chicago (Chicago, IL) - University of Texas Health Science Center at Houston (Houston, TX) Data analysis included Fisher exact tests to compare categorical variables across COVID test groups and Wilcoxon rank sum tests to compare continuous covariates. Two-sample tests of proportions were used to compare observed COVID-19 positivity rates to other study results. RESULTS: Out of 684 patients in 3 institutions, 17 patients (2.5%, 95% CI = 1.5 to 4.0%) tested positive for COVID-19 over a 10 month interval (March 1, 2020- December 31, 2020). The majority of patients that tested positive were asymptomatic in the preoperative setting (P-value = .09). They were significantly more likely to be African-American (P-value = .015) and less likely to have a stay-at-home order present at the time of surgery (P-value = .033). Age, gender, and hospital location did not play a statistically significant role. CONCLUSION: Our results demonstrate a 2.5% incidence of COVID-19 infection in the total population of patients undergoing scheduled oral-maxillofacial surgeries in 3 major healthcare systems across the United States. This data may help inform perioperative protocols and infection control measures during the COVID-19 pandemic.

Biomaterials-aided mandibular reconstruction using in vivo bioreactors.

Large mandibular defects are clinically challenging to reconstruct due to the complex anatomy of the jaw and the limited availability of appropriate tissue for repair. We envision leveraging current advances in fabrication and biomaterials to create implantable devices that generate bone within the patients themselves suitable for their own specific anatomical pathology. The in vivo bioreactor strategy facilitates the generation of large autologous vascularized bony tissue of customized geometry without the addition of exogenous growth factors or cells. To translate this technology, we investigated its success in reconstructing a mandibular defect of physiologically relevant size in sheep. We fabricated and implanted 3D-printed in vivo bioreactors against rib periosteum and utilized biomaterial-based space maintenance to preserve the native anatomical mandibular structure in the defect site before reconstruction. Nine weeks after bioreactor implantation, the ovine mandibles were repaired with the autologous bony tissue generated from the in vivo bioreactors. We evaluated tissues generated in bioreactors by radiographic, histological, mechanical, and biomolecular assays and repaired mandibles by radiographic and histological assays. Biomaterial-aided mandibular reconstruction was successful in a large superior marginal defect in five of six (83%) sheep. Given that these studies utilized clinically available biomaterials, such as bone cement and ceramic particles, this strategy is designed for rapid human translation to improve outcomes in patients with large mandibular defects.

Comprehensive Oromandibular Reconstruction Utilizing an Anterolateral Thigh Free Tissue Transfer Flap and Tissue Engineering: Technical and Timing Considerations.

The present report describes the technical nuances involved in oromandibular reconstruction utilizing a soft tissue free flap and tissue engineering in a step wise fashion for complete oral rehabilitation.

Is Reconstruction of Large Mandibular Defects Using Bioengineering Materials Effective?

PURPOSE: Clinical tissue engineering has revolutionized surgery by improving surgical efficiency and decreasing the risks associated with traditional bone graft procurement techniques. Compared with autogenous bone grafts, composite tissue-engineered grafts fulfill the principles of osteoconduction, osteoinduction, and osteogenesis and provide adequate bone volume for maxillofacial reconstruction with less morbidity. The present study aimed to demonstrate the effectiveness, as defined by our success criteria, of a composite tissue-engineered bone graft in the reconstruction of mandibular defects. PATIENTS AND METHODS: We implemented a retrospective case series and enrolled a sample of patients with mandibular defects that had been reconstructed using allogeneic bone combined with recombinant human bone morphogenic protein-2 and bone marrow aspirate concentrate at our institution during a 5-year period. The success criteria were as follows: 1) bone union, defined as a homogenous radiopaque pattern continuous with native bone without mandibular mobility; and 2) volume of grafted bone adequate for implant placement, defined as at least 1.0 cm (height) by 0.8 cm (width). Clinical examinations and computed tomography scans were performed at 6 months postoperatively. Descriptive statistics were computed for each variable. RESULTS: From 2014 to 2019, tissue engineering reconstruction was used in 31 patients with and 3 patients without mandibular continuity defects, for a total of 34 patients. The median follow-up was 6 months. The mean length of the continuity defects was 5.5 cm (range, 1.0 to 12.5). Of the 30 patients with mandibular continuity defects, 27 achieved success according to our criteria, with an average gained height of 2.12 ± 0.64 cm and width of 1.53 ± 0.46 cm. Of the 34 patients, 1 was lost to follow-up, and treatment failed in 3 patients. CONCLUSIONS: Although the use of autogenous graft remains the reference standard, the evolving science behind clinical tissue engineering has resulted in an effective treatment modality for complex head and neck defects with less morbidity and graft material equal to that of autogenous bone.

Palatal Basal Cell Adenocarcinoma From Misdiagnosis to Cure: A Unique Case Report and Review.

We describe a 66-year-old woman who received a misdiagnosis of benign neoplasms twice before receiving the appropriate diagnosis of basal cell adenocarcinoma. At the time of recurrence, her care was assumed by the senior author, who made the appropriate diagnosis and administered treatment including transoral microvascular reconstruction, as well as adjuvant radiation therapy, which achieved a cure. A review of the current literature on this disease entity also is included.

Single-Unit 3D-Printed Titanium Reconstruction Plate for Maxillary Reconstruction: The Evolution of Surgical Reconstruction for Maxillary Defects-A Case Report and Review of Current Techniques.

The complex 3-dimensional anatomy of the facial skeleton creates a unique challenge for surgical reconstruction. Even more difficult is the precise reconstruction of the maxilla owing to its lack of solid bony support after large resections. Virtual surgical planning (VSP) technology has many applications in head and neck surgery, such as preoperative planning, fabrication of cutting guides and stereolithographic models, and fabrication of custom implants. We present the case of a patient who had undergone surgical resection of a mucoepidermoid carcinoma of the maxilla and immediate reconstruction with a vascularized free fibula flap using VSP. Using a custom 3-dimensional, titanium printed plate, which corresponded precisely with the surgical defect, the maxilla and midface were reconstructed to ideal dimensions with no unplanned surgical manipulation and a shorter overall operating time. We have described the technique and reviewed the pertinent reported data.

Massive Self-Induced Subcutaneous Cervicofacial, Pneumomediastinum, and Pneumopericardium Emphysema Sequelae to a Nondisplaced Maxillary Wall Fracture: A Case Report and Literature Review.

Iatrogenic cervicofacial subcutaneous emphysema is a well-reported entity in the field of dentistry and oral and maxillofacial surgery, especially with the use of air-driven headpieces. Cervicofacial subcutaneous emphysema sequelae after maxillofacial trauma, however, has been reported less and the self-induced variant is even rarer. We report a case of massive cervicofacial subcutaneous emphysema, pneumomediastinum, and pneumopericardium in a healthy 16-year-old boy after blunt trauma to the face, which caused a nondisplaced anterior maxillary wall fracture. The findings from the present case report will validate the common phrase "no nose blowing or holding your sneezes" that clinicians tell patients after maxillofacial trauma and sinus surgery.

Initial Manifestation of Acquired Hemophilia A After a Routine Tooth Extraction. A Case Report and Literature Review.

Although surgical treatment of patients on anticoagulation regimens is common practice among oral and maxillofacial surgeons, unexpected and unknown coagulopathies can have devastating and catastrophic consequences for the most routine of procedures. Acquired hemophilia A (AHA) is an extremely rare life-threatening bleeding disorder characterized by autoantibodies directed against circulating coagulation factor VIII. The effects of AHA can produce catastrophic bleeding and hematomas. The effect of this uncontrolled hemorrhage after dentoalveolar surgery can mimic severe head and neck infection by causing dysphagia, odynophagia, and acute airway complications. This report describes the case of a 64-year-old woman who was diagnosed with AHA after routine extraction of the mandibular left third molar.

Unusual Case of a Massive Macroglossia Secondary to Myxedema: A Case Report and Literature Review.

Macroglossia is classified as true macroglossia, which exhibits abnormal histology with clinical findings, and relative macroglossia, in which normal histology does not correlate with pathologic enlargement. This report describes an atypical case of morbidity with massive macroglossia secondary to myxedema; the macroglossia enlarged over a 3-month period before being presented to the Department of Oral and Maxillofacial Surgery, University of Texas Health Science Center at Houston (Houston, TX). Substantial enlargement of the tongue (16 cm long × 10 cm wide) was first attributed to angioedema, which was refractory to the discontinuation of lisinopril and a C1 esterase inhibitor. A core tongue biopsy examination was performed to rule out angioedema, amyloidosis, myxedema, and idiopathic muscular hypertrophy. Interstitial tissue was positive for Alcian blue and weakly positive for colloidal iron, which are correlated with hypothyroidism and a diagnosis of myxedema. However, the macroglossia did not resolve after correcting for hypothyroidism. The patient required a wedge glossectomy for definitive treatment. She recovered unremarkably, with excellent cosmesis and preservation of lingual and hypoglossal function. There are some case reports of massive macroglossia but none with myxedema as the primary etiology.

The Use of Acellular Urinary Bladder Matrix as Coverage for Fasciocutaneous Free Flap Donor Sites: An Alternative to Traditional Grafting Procedures.

PURPOSE: The purpose of this review is to 1) evaluate our early experience with urinary bladder matrix as a dressing for the management of complex wounds subsequent to fasciocutaneous or osteocutaneous vascularized tissue harvests, 2) assess coverage of exposed tendons and the duration of granulation and epithelial coverage, and 3) assess esthetic and functional outcomes compared with traditional means for the management of vascularized tissue harvest sites. PATIENTS AND METHODS: This prospective study consisted of 8 consecutive patients in whom a substantial cutaneous component was harvested as part of a vascularized tissue reconstruction, which did not permit for primary closure (n = 7) or had skin graft failure (exposed tendon) that required additional treatment (n = 1). RESULTS: Functional and esthetic outcomes were comparable to those of traditional methods of donor site closure as determined by clinical evaluation and subjective assessment by the patient. In all patients, no additional grafting was required to achieve full coverage. Irregularities of the wound achieved a level plane with granulation tissue an average of 3 to 4 weeks after surgery, followed by an additional 5 to 8 weeks for epithelialization. Wound care was minimal and included twice-daily wet-to-dry dressings. It was noted that complete wound healing was delayed in the urinary bladder matrix group, which required more attention during wound care when compared with published data. Advantages included facilitation of a robust granulation layer that leveled wound irregularities and avoidance of an additional skin graft site. CONCLUSIONS: The use of urinary bladder matrix is a viable option for the rehabilitation of donor sites of vascularized tissue that include skin. Benefits include the avoidance of an additional skin graft donor site, facilitation of epithelialization over exposed tendons, leveling of the donor site texture, and an equivalent esthetic result compared with current practices for wound coverage. The success of this technique may be limited by the cost of materials and the protracted course for wound epithelialization.

Reconstruction of a Post-Traumatic Maxillary Ridge Using a Radial Forearm Free Flap and Immediate Tissue Engineering (Bone Morphogenetic Protein, Bone Marrow Aspirate Concentrate, and Cortical-Cancellous Bone): Case Report.

The purpose of this article is to describe reconstruction of the maxillary alveolar ridge by use of a microvascular free flap combined with an immediate tissue-engineered bone graft. This novel surgical technique involved the use of a radial forearm free flap and immediate allogeneic avascular bone graft augmented with bone morphogenetic protein and bone marrow aspirate concentrate. A poly-d,l-lactic acid mesh was used as a containment unit for the bone graft. The patient was successfully treated with a viable radial forearm free flap for soft tissue and regeneration of bone with adequate height and width, which allowed the placement of 3 dental implants with excellent arch coordination. We believe this is the first published case describing such a technique to reconstruct the maxillary alveolus.

Regeneration of a Tooth in a Tissue-Engineered Mandible After Resection of a Central Giant Cell Tumor. Demonstrating Evidence of Functional Matrix Theory and Ectodermal Origin of Teeth in a Human Model-A Case Report.

Central giant cell tumors (CGCTs) are uncommon lesions occurring in the jaw. They are benign but locally destructive osteolytic lesions. They usually occur in pediatric patients 5 to 15 years of age. Multiple noninvasive modalities of treatment (intralesional steroids, interferon, calcitonin, and denosumab) have been described for those lesions, but for those that are refractory to treatment, enucleation and curettage or resection is a curative surgery. This case report describes a pediatric patient who was diagnosed with an aggressive CGCT of the left mandible encompassing the right angle to the condyle. The lesion became refractory to noninvasive treatments and immediate resection and reconstruction was performed using principles of tissue engineering. After 5 years of close observation, the patient showed normal morphology and growth of his mandible, but surprisingly developed a left mandibular third molar (tooth 17) in the site of the mandibular resection and reconstruction. This is the first case report in the literature to show the spontaneous development of teeth in a human reconstructed mandible, contributing evidence toward the functional matrix theory of mandibular growth and ectodermal origin of teeth.

Immediate Transoral Allogeneic Bone Grafting for Large Mandibular Defects. Less Morbidity, More Bone. A Paradigm in Benign Tumor Mandibular Reconstruction?

PURPOSE: Reconstruction of hard tissue continuity defects caused by ablative tumor surgery has been traditionally reconstructed with autogenous bone grafts or microvascular free flaps. Although results have been predictable from these 2 methods of reconstruction, the morbidity associated with bone harvest is quite serious for the patient. Predictable results have been obtained with using a combination of 100% cadaver bone, bone marrow aspirate concentrate (BMAC), and recombinant human bone morphogenic protein in immediate reconstruction for benign tumor extirpations through the extraoral approach. In light of these successful outcomes, the same combination was evaluated with an intraoral approach. This study evaluated the success of immediate mandibular reconstruction through the intraoral approach without any autogenous bone harvesting. PATIENTS AND METHODS: The aim of this retrospective study was to share the authors' experience with the use of 100% allogeneic bone in combination with bone morphogenic protein and BMAC through the transoral approach for immediate reconstruction of continuity defects that resulted from benign tumor surgery. A retrospective chart review was performed of all patients undergoing bone graft reconstruction at the University of Texas Health Sciences Center at Houston (UTHealth) Department of Oral and Maxillofacial Surgery from December 2014 through January 2016. Inclusion criteria were biopsy-proven benign tumors, American Society of Anesthesiologists I or II health status, and adequate intraoral soft tissue for primary closure determined during initial consultation. RESULTS: Five patients who underwent this procedure at the UTHealth Department of Oral and Maxillofacial Surgery from December 2014 through January 2016 are presented. The success rate was 100%. All patients showed excellent bone quality clinically and radiographically for endosseous dental implant placement. With the transoral approach and no autogenous bone harvesting, the average operating time was 3.4 hours and the hospital stay was 2.4 days. CONCLUSIONS: Composite allogeneic tissue engineering is an effective and predictable technique for immediate reconstruction of continuity defects from ablative benign tumor surgery. Overall, there was no donor site morbidity, the intraoperative time was shorter, there were fewer admission days, and total costs overall were lower compared with traditional methods.

Reconstruction of Temporal Hollowing Defect With Anterior-Lateral Thigh Free Flap Following Resection of Recurrent Ameloblastoma of the Infratemporal Fossa and Right Mandible (a Case Report).

Recurrent invasive ameloblastoma of the infratemporal fossa is an uncommonly encountered phenomenon in the practice of oral and maxillofacial surgery and presents many surgical challenges for the practitioner. This case report describes a patient who underwent previous resection of a mandibular ameloblastoma with multiple recurrences. The patient was diagnosed with a recurrent ameloblastoma of the infratemporal fossa that was subsequently resected and reconstructed using an anterolateral thigh (ALT) free tissue transfer. There are few reported cases of recurrent ameloblastomas in the infratemporal fossa and none that describe surgical resection and reconstruction of such a lesion. Owing to the uniqueness of the surgical defect, an ALT flap was used to correct the temporal hollowing. There have been multiple reported cases of reconstruction of temporal hollowing defects using autogenous fat or allograft; however, none have described the use of a de-epithelialized ALT microvascular reconstruction of a temporal hollowing defect. This case report describes a unique clinical situation of surgical resection and reconstruction that resulted in a satisfactory outcome for the patient.

Use of Botox (OnabotulinumtoxinA) for the Treatment of Parotid Sialocele and Fistula After Extirpation of Buccal Squamous Cell Carcinoma With Immediate Reconstruction Using Microvascular Free Flap: A Report of 3 Cases.

PURPOSE: Buccal squamous cell carcinoma is an aggressive form of oral carcinoma with a high recurrence rate. Injury to the parotid duct is often unavoidable when surgically treating buccal squamous cell carcinoma because of the intimate anatomic relation among the buccal mucosa, Stensen duct, and parotid gland. It is often difficult to achieve negative margins and preserve the integrity of the parotid duct. Sialocele formation is a frequent and untoward complication owing to extravasation of saliva into the surgical defect, which delays healing, creates fistulas, and produces painful facial swelling. Currently, no consensus exists regarding the management of a parotid sialocele. Multiple investigators have described different modalities of treatment, such as repeated percutaneous needle aspiration, pressure dressings, antisialagogue therapy, radiotherapy, botulinum toxin, and surgical techniques, including duct repair, diversion, ligation, drain placement, and parotidectomy. MATERIALS AND METHODS: With approval from the institutional review board of the University of Texas Health Sciences Center at Houston, 3 cases of parotid sialocele and nonhealing fistulas successfully treated with Botox (onabotulinumtoxinA) after tumor extirpation, neck dissection, and reconstruction with a microvascular free flap are presented. RESULTS: At the University of Texas Health Sciences Center at Houston, the radiation oncologist prefers not to start adjunctive radiation treatment with a nonhealing wound or a drain in the field of radiation. Ideally, a standard timing of adjuvant radiotherapy is 6 to 8 weeks after surgery and 60 cGy should be completed before 7 months. CONCLUSIONS: With the use of Botox, the nonhealing wound resolved and the drain was removed at least 2 weeks before the initiation of adjunctive radiotherapy, thus minimizing the delay in adjuvant treatment.

Are Patients with Syndromic Craniosynostosis at Greater Risk for Epilepsy than Patients with Nonsyndromic Craniosynostosis?

OBJECTIVE: The aim of this study was to determine whether patients with syndromic craniosynostosis (SCS) are at increased risk for epilepsy relative to patients with nonsyndromic craniosynostosis (NSCS). METHODS: A retrospective cohort study was completed using the Kids' Inpatient Database (KID). All patients diagnosed with craniosynostosis (CS) were included. The primary predictor variable was study grouping (SCS vs. NSCS). The primary outcome variable was a diagnosis of epilepsy. Descriptive statistics, univariate analyses and multivariate logistic regression were performed to identify independent risk factors for epilepsy. RESULTS: The final study sample included a total of 10,089 patients (mean age, 1.78 years ± 3.70; 37.7% female). 9278 patients (92.0%) had NSCS, and the remaining 811 patients (8.0%) had SCS. A total of 577 patients (5.7%) had epilepsy. Not controlling for other variables, patients with SCS were at increased risk for epilepsy relative to patients with NSCS (OR 2.1, P < 0.001). After controlling for all significant variables, patients with SCS were no longer at increased risk for epilepsy relative to patients with NSCS (OR 0.73, P = 0.063). Hydrocephalus, Chiari malformation (CM), obstructive sleep apnea (OSA), atrial septal defect (ASD), gastro-esophageal reflux disease (GERD) were all independent risk factors (P < 0.05) for epilepsy. CONCLUSIONS: Syndromic craniosynostosis (SCS) in itself is not a risk factor for epilepsy relative to NSCS. The greater prevalence of hydrocephalus, CM, OSA, ASD, and GERD, all of which were risk factors for epilepsy, in patients with SCS relative to patients with NSCS likely explains the greater prevalence of epilepsy in SCS relative to NSCS.

Artificial intelligence-based epigenomic, transcriptomic and histologic signatures of tobacco use in oral squamous cell carcinoma.

Oral squamous cell carcinoma (OSCC) biomarker studies rarely employ multi-omic biomarker strategies and pertinent clinicopathologic characteristics to predict mortality. In this study we determine for the first time a combined epigenetic, gene expression, and histology signature that differentiates between patients with different tobacco use history (heavy tobacco use with ≥10 pack years vs. no tobacco use). Using The Cancer Genome Atlas (TCGA) cohort (n = 257) and an internal cohort (n = 40), we identify 3 epigenetic markers (GPR15, GNG12, GDNF) and 13 expression markers (IGHA2, SCG5, RPL3L, NTRK1, CD96, BMP6, TFPI2, EFEMP2, RYR3, DMTN, GPD2, BAALC, and FMO3), which are dysregulated in OSCC patients who were never smokers vs. those who have a ≥ 10 pack year history. While mortality risk prediction based on smoking status and clinicopathologic covariates alone is inaccurate (c-statistic = 0.57), the combined epigenetic/expression and histologic signature has a c-statistic = 0.9409 in predicting 5-year mortality in OSCC patients.