• Journal of the Korean Association of Oral and Maxillofacial Surgeons
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• v.47 no.3
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• pp.209-215
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• 2021

Objectives: Vascular malformation (VM) of the tongue can cause true macroglossia in children. Reduction glossectomy provides primary relief when sclerotherapy has failed or is not possible. In this study, we evaluated the surgical role in functional outcome of reduction glossectomy performed for VM of the tongue. Patients and Methods: We evaluated the functional and surgical outcomes of seven children who were treated at a tertiary care centre in Southern India between 2013 and 2018. Results: Six children underwent median glossectomy, while one child underwent lateral glossectomy. Functional assessment was performed at least 2 years after the date of surgery. At the time of assessment, speech was comprehensible for three children and was occasionally unintelligible in four children. Taste and swallowing were normal in all seven children. Six children exhibited a minimal residual lesion after surgery, of which only one was symptomatic. Residual lesions were managed with sclerotherapy (n=3), observation (n=2), or repeat surgery (n=1). Conclusion: Reduction glossectomy in children with macroglossia secondary to VMs has acceptable outcomes in terms of cosmesis and speech, with no gastronomic restriction.

• Clinical and Experimental Pediatrics
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• v.53 no.3
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• pp.432-436
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• 2010

Transient neonatal diabetes mellitus (TNDM) has been associated with paternal uniparental isodisomy of chromosome 6, paternally inherited duplication of 6q24, or a methylation defect at a CpG island of the ZAC or HYMAI gene. We experienced a case of TNDM in which the patient presented with hyperglycemia, macroglossia, and intrauterine growth retardation, caused by a paternally derived HYMAI. An 18-day-old female infant was admitted to the hospital because of macroglossia and recurrent hyperglycemia. In addition to the macroglossia, she also presented with large fontanelles, micrognathia, and prominent eyes. Serum glucose levels were 200-00 mg/dL and they improved spontaneously 2 days after admission. To identify the presence of a maternal methylated allele, bisulfite-treated genomic DNA from peripheral blood was prepared and digested with BssHII after polymerase chain reaction (PCR) amplification with methylation-specific HYMAI primers. PCR and restriction fragment length polymorphism analysis showed that the patient had only the paternal origin of the HYMA1 gene. TNDM is associated with a methylation defect in chromosome 6, suggesting that an imprinted gene on chromosome 6 is responsible for this phenotype.

• Maxillofacial Plastic and Reconstructive Surgery
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• v.36 no.5
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• pp.214-218
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• 2014

Macroglossia can create dental and skeletal instability after orthodontic treatment or orthognathic surgery for mandibular prognathism. In relevant literature, partial glossectomy is suggested for a good post-treatment prognosis. Most of the published partial glossectomy cases are two-staged surgery, because of concern about postoperative airway obstruction. As orthognathic surgical techniques and fixation method develop, however, concerns about postoperative airway obstruction have lessened. In this case, mandibular setback surgery and partial glossectomy were performed simultaneously, leading to stable recovery without any postoperative respiratory problems. After surgical technique to preserve the tongue tip, we achieved good outcomes without postoperative side effects of lingual hypoesthesia, pronunciation disorder and dyskinesia. We report this case with a literature review.

• The Journal of Korea Assosiation for Disability and Oral Health
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• v.12 no.2
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• pp.92-95
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• 2016

Beckwith-Wiedemann syndrome comprises multiple congenital anomalies with a risk of development of childhood tumors. Macroglossia is the predominent finding in Beckwith-Wiedemann syndrome and other findings include postnatal somatic gigantism, abdominal wall defects, nevus flammeus of the face, congenital heart defects, and cleft palate. The aim of this case presentation is to describe the dental treatment of a child with Beckwith-Wiedemann syndrome. A 3-year-old boy with Beckwith-Wiedemann syndrome visited to the Seoul National University Dental Hospital for dental evaluation and treatment. Due to the multiple caries and poor cooperation, we planned to perform the dental procedure under general anesthesia. The dental procedure was successfully performed and there was no airway complications after extubation. This case suggests that general anesthesia may be useful for the patient with uncooperative Beckwith-Wiedemann syndrome, since macroglossia and laryngomalacia may exacerbates airway compromise during dental sedation.

• Journal of Dental Rehabilitation and Applied Science
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• v.34 no.3
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• pp.218-224
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• 2018

Macroglossia is an obstacle in regard to general prosthodontic restoration. Not only is it difficult to obtain support and stability from dentures manufactured from normal protocol, obtaining efficacy from basic mastication is also difficult. In such cases, realizing harmony between the occlusion and surrounding muscle structures may be important with regard to manufacture of stable full dentures, and it is necessary to form the appropriate polished surface for this case. The neutral zone is defined as the potential area resulting from the neuromuscular function that results in equilibrium between the outward force exerted by the tongue and the inward force exerted by the lips and cheeks. The artificial teeth of the full denture lies in this area, and if the polished zone follows the anatomic form of the dynamic muscles, the movement of the muscles simply acts as an element of stabilizing the dentures improving the esthetics through facial support.

• The korean journal of orthodontics
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• v.5 no.1
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• pp.69-73
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• 1975

This is case report of true class III malocclusion with macroglossia is corrected by glossectomy in 13 years female patient. After orthodontic treatment, the patient is bound to glossectomy because the corrected condition is relapsed to the previous condition due to relatively enlarged tongue compared with the original dental arch. By the interpretation of the cephalogram and model analysis, it is approved that the growth pattern and direction are normal range and mandible is located anterioly to the cranium. The results are follows: 1. We could treat the true Cl III malocclusion. 2. We could prevent the relapse of the treated condition by the surgical intervention, such as partial glossectomy. 3. Sensory, speech, swallowing and so other functions after the operation have been with in normal limit without any serious complications or seguellae.

• Sleep Medicine and Psychophysiology
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• v.12 no.2
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• pp.105-110
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• 2005

The pathogenesis and mechanism of obstructive sleep apnea (OSA) has been under investigation for over 25 years, but its etiology and mechanism remains elusive. Skeletal (maxillary and/or mandibular hypoplasia or retrodisplacement, inferior displacement of hyoid) and soft tissue (increased volume of soft tissue, adenotonsillar hypertrophy, macroglossia, thickened lateral pharyngeal walls) factors, pharyngeal compliance (increased), pharyngeal muscle factors (impaired strength and endurance of pharyngeal dilators and fixators), sensory factors (impaired mechanoreceptor sensitivity, impaired pharyngeal dilator reflexes), respiratory control system factors (unstable respiratory control) and so on facilitate collapse upper airway. Therefore, OSA may be a heterogeneous disorder, rather than a single disease entity and various pathogenic factors contribute to the OSA varies person to person. As a result, patients may respond to different therapeutic approaches based on the predominant abnormality leading to the sleep-disordered breathing.

• Advances in pediatric surgery
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• v.6 no.2
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• pp.160-165
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• 2000

Beckwith-Wiedemann syndrome presents with multisystemic patterns of congenital anomalies and macrosomia. This syndrome was independently described by Beckwith in 1963 and by Wiedemann in 1964. There is wide spectrum of clinical manifestations, including prenatal or postnatal overgrowth, neonatal hypoglycemia, macroglossia, visceromegaly, omphalocele, hemihypertrophy and a predisposition for embryonal tumors, most frequently Wilms' tumor. We managed a case of Beckwith-Wiedemann syndrome with left adrenal cortical neoplasm of undetermined malignancy.

• Journal of Dental Anesthesia and Pain Medicine
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• v.18 no.2
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• pp.115-117
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• 2018

Lingual nerve injury is a rare complication of general anesthesia. The causes of lingual nerve injury following general anesthesia are multifactorial; possible mechanisms may include difficult laryngoscopy, prolonged anterior mandibular displacement, improper placement of the oropharyngeal airway, macroglossia and tongue compression. In this report, we have described a case of bilateral lingual nerve injury that was associated with orotracheal intubation for open reduction and internal fixation of the left distal radius fracture in a 61-year-old woman. In this case, early treatment with dexamethasone effectively aided the recovery of the injured lingual nerve.

• Sleep Medicine and Psychophysiology
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• v.5 no.2
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• pp.210-215
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• 1998

OSA(Obstructive Sleep Apnea) is a condition with repeated obstruction of the upper airway while sleeping. This obstruction of the upper airway may result if sleep-induced physiologic change in muscle activity is superimposed on various structural defects of the upper airway. Macroglossia, micrognathia, retrognathia, and decreased airway lumen have all been implicated in OSA. There are a variety of surgical and nonsurgical treatment modalities currently available for OSA. Recently the uvulopalatopharyngoplasty has achieved widespread use, but success rates vary and long-term effects are still unknown. Current nonsurgical treatment methods can be cumbersome and uncomfortable with variable responses. For this case. we diagnosed as OSA by using polysomnographic analysis and cephalometric analysis, and delivered intraoral removable appliance which is more conservative, reversible and easy to use for patient. And we got results that Apnea Index(AI) decreased from 14.4 to 3.1, Total Respiratory Disturbance Index(Total RDI) decreased from 18.1 to 7.0 and removal of chronic headache in daytime.