What is Idiopathic Macroglossia? A case series and review of literature

Purpose Macroglossia is pathological condition which resulted in hypertrophy of tongue muscles. It is classified into congenital and acquired macroglossia. Idiopathic macroglossia (IM) is tongue hypertrophy without systemic or genetic causes. This condition has a significant impact on the quality of life, and often require tracheostomy and percutaneous endoscopic gastrostomy (PEG) to sustain living. Guidelines regarding treatment for this subtype are scant. The purpose of this project to present diagnosis and management of a series of patients with idiopathic macroglossia. Methods This was a retrospective case series of a cohort of patients with IM who were treated by Oral and Maxillofacial Surgery (OMS) service at University of Texas Health Science Center in Houston (UTHealth) and Emory University from 2019 to 2021. Inclusion criteria are (1) 18 years or older, (2) diagnosed with macroglossia, (3) managed with surgery, (4) with normal tongue tissue on histopathology results, and (5) with a negative COVID test. Patients were excluded if they are younger than 18 years old and diagnosed with macroglossia due to an underlying etiology such as congenital anomaly, systemic conditions, and intraoral inflammatory changes. Study variables were patient demographics, social history, medical comorbidities, clinical presentation, clinical dimensions, presence of tongue protrusion, difficulty feeding, difficulty in breathing, imaging characteristics/dimensions, pathological findings, management (tracheostomy, PEG, glossectomy), and length of inpatient stay. The outcome variables were normalization of tongue size, return of parenteral nutrition, and able to tolerate tracheostomy decannulation. Data were collected using a standardized collection form. Descriptive statistics were computed. Results Five patients (mean age, 45 years) with IM met inclusion criteria. All patients had history of hypertension, cerebral vascular disease, and prolong intubation. All patients presented with anterior tongue enlargement, with mean dimension of 13 × 6 cm, full or partial dentition, altered tongue sensation. They were all managed with tracheostomy to secure the airway, PEG and partial glossectomy. Average length of inpatients stay was 10 days. All IM achieved clinical resolution, 80% of the patients had their tracheostomy decannulated and PEG tube removed. Conclusion Management of macroglossia requires multidisciplinary approach. While etiology can often be identified and medical treatment can be initiated for reversible causes, most of the macroglossia cases previously reported required surgical management. Surgical reduction offers the best functional and cosmetic results and minimizes morbidity. In the case idiopathic macroglossia, management should involve tracheostomy and feeding access for the initial stabilization followed by reduction glossectomy for improvement of functional outcomes.

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.

Massive macroglossia, a rare side effect of COVID-19: clinical, histologic, and genomic findings in COVID-19-positive versus COVID-19-negative patients.

PURPOSE: The primary purpose of this study is to identify if there is an underlying genetic predisposition for COVID-related macroglossia and if this susceptibility is higher among individuals of African heritage. Secondary objectives include determining if genetic testing of COVID-infected patients who are intubated and prone could identify patients with higher susceptibility to the development of macroglossia. METHODS: A retrospective chart review was completed for each patient, and prospectively, genetic and histopathologic analyses were completed. Whole-exome sequencing was completed on two patients; immunohistochemistry was completed on the COVID-positive tissue samples. RESULTS: Histopathology of the COVID-positive patient revealed significant peri-lymphocytic infiltrate, which was absent in the COVID-negative patient. Immunohistochemistry confirmed the presence of immune cells. Results from the whole-exome sequencing were inconclusive. CONCLUSION: The findings of this study are consistent with others that have observed a lymphocytic infiltrate in the organs of patients infected with SARS-CoV-2. On histology, IHC highlighted a CD45 + predominance, indicating that a robust immune response is present in the tissues. The pathobiology of this phenomenon and its role in the development and/or persistence of massive macroglossia requires further study.