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At least 271 records · Page 15Linked to original sources
PubMed1996

Anesthetic considerations of two sisters with Beckwith-Wiedemann syndrome.

Anesthetic considerations of 21-mo-old and 4-yr-old sisters with Beckwith-Wiedemann syndrome during surgical repair of cleft palate and reduction of macroglossia are presented and discussed. This syndrome is characterized by exomphalos, macroglossia, gigantism, hypoglycemia in infancy, and many other clinical features. This syndrome is also known as exomphalos, macroglossia, and gigantism (EMG) syndrome. Principal problems associated with anesthetic management in this syndrome are hypoglycemia and macroglossia. Careful intraoperative plasma glucose monitoring is particularly important to prevent the neurologic sequelae of unrecognized hypoglycemia. It is expected that airway management would be complicated by the macroglossia, which might cause difficult bag/mask ventilation and endotracheal intubation following the induction of anesthesia and muscle paralysis, so preparations for airway difficulty (e.g., awake vocal cord inspection) should be considered before induction. A nasopharyngeal airway is useful in relieving postoperative airway obstruction.

Airway Obstruction↗
PubMed1984

Macroglossal lymphangioendotheliomatosis. Report of a case in a pregnant woman.

An unusual case of adult-onset lymphangioendotheliomatous macroglossia occurred in a 37-year-old woman. In addition to a dramatic acceleration of the macroglossia during her third pregnancy, the patient exacerbated the macroglossia by Valsalva's maneuvers and voluntary tongue protrusion. Bed rest and head elevation could improve the macroglossia within 24 hours. Preservation of an adequate airway during a life-threatening state of proliferation and expansion of the lymphangiomatous lesions required surgical intervention. The patient's lesion differed clinically and pathologically from usual glossal lymphangioma, hemangioma, pregnancy gingival tumor, and hemangiomatosis. Pathologically, multifocal, multicentric areas of cavernous hemangiomatouslike, cavernous lymphangiomatouslike, and angioendotheliomatous areas were present within the deep musculature of the tongue, minor salivary tissue, and soft tissue of the floor of the mouth.

Adult↗
PubMed2000

Tongue reduction in Beckwith-Weidemann syndrome.

OBJECTIVE: To review our experience with patients with macroglossia as a component of Beckwith-Weidemann Syndrome (BWS). DESIGN: Chart review of six patients treated with BWS. SETTING: Tertiary care teaching hospital. PATIENTS: Six patients diagnosed with BWS and macroglossia. INTERVENTIONS: Four patients underwent at least one surgical procedure to address their macroglossia. The surgical options and potential complications are discussed. RESULTS: Three patients who have undergone tongue reduction have a functioning tongue with normal mobility. Two patients have required tracheotomy as apart of their management and still have significant tongue enlargement. CONCLUSIONS: Macroglossia as a part of BWS may present a difficult management problem. Various methods of tongue reductions have been reported with mixed results.

Beckwith-Wiedemann Syndrome↗
PubMed1999

Effects of surgical reduction of the tongue.

There are still no objective diagnostic criteria for macroglossia. The aims of this study were to examine the effect of reduction of the tongue on its position, and to suggest a standard measurement point for the objective diagnosis of macroglossia. Twenty-four patients were studied. Lateral cephalometric radiographs were taken with the tongue in the rest position, and preoperative and postoperative measurements were made on cephalometric lateral roentgenograms from all 14 patients with macroglossia and the 10 control subjects. The results suggested that measurement of length of the tongue, and the tongue area: oral cavity ratio on lateral cephalometric radiographs are useful in the more accurate diagnosis of macroglossia.

Adult↗
PubMed1995

Head and neck manifestations of Beckwith-Wiedemann syndrome.

Beckwith-Wiedemann syndrome is a congenital disorder manifested by organomegaly, omphalocele, hypoglycemia, and macroglossia. We have found a significant number of these children to be at risk for upper airway obstruction during infancy or childhood. In this review of 13 children, 2 required tracheotomy during infancy for cor pulmonale caused by macroglossia. Seven of nine children older than 1 year required tonsillectomy and adenoidectomy to relieve upper airway obstruction. Although macroglossia can be a cause of airway obstruction in infants with Beckwith-Wiedemann syndrome, we have found that airway obstruction during childhood is related to tonsillar and adenoidal hypertrophy and not to macroglossia. Anterior tongue reduction is reserved for the correction of malocclusion, articulation errors, or cosmesis, whereas tonsillectomy and adenoidectomy may be curative of obstructive symptoms.

Adenoidectomy↗
PubMed2002

A less-invasive approach with orthodontic treatment in Beckwith-Wiedemann patients.

The Beckwith-Wiedemann syndrome (BWS) is a rare genetic disorder, linked to an alteration on the short arm of chromosome 11 that comprises multiple congenital anomalies. Macroglossia is the predominant finding, with subsequent protrusion of dentoalveolar structures, which results in a protruding mandible, anterior open bite, abnormally obtuse gonial angle and increased mandibular length. A less-invasive treatment with orthopaedic appliances in a patient with early tongue reduction is presented. This work summarizes the oral signs linked to macroglossia, and highlights the influence of macroglossia on mandibular growth structures. In our opinion, glossotomy could be carried out in the paediatric patient as a preventive measure in that it curbs the tongue's influence on skeletal growth and dramatically reduces the duration and extensiveness of subsequent treatment.

Age Factors↗
PubMed1995

Treatment of the craniofacial complications of Beckwith-Wiedemann syndrome.

Variable degrees of macroglossia have been noted in patients with Beckwith-Wiedemann syndrome. Past studies have shown that a major effect of the macroglossia is protrusion of dentoalveolar structures, resulting in an anterior open bite and a prognathic mandibular appearance secondary to an abnormally obtuse gonial angle and increased effective mandibular length. In our series of 11 Beckwith-Wiedemann syndrome patients we have observed that early correction of the macroglossia by means of partial glossectomy has resulted in decreased anterior open bite and mandibular prognathism as compared with patients managed conservatively.

Beckwith-Wiedemann Syndrome↗