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Diagnosis of macroglossia and indications for reduction glossectomy
ORIGINAL ARTICLES
Diagnosis of macroglossia and indications for reduction glossectomy Larry M. Wolford, DDS," and David A. Cottrell, DMD b Dallas, Texas, and Boston, Mass. Macroglossia can create dentomusculoskeletal deformities, instability of orthodontic and orthognathic surgical procedures, and masticatory, speech, and airway problems. The cause of macroglossia must be clearly defined, and true macroglossia separated from pseudomacroglossia (displacement of the tongue created by anatomic factors other than tongue size alone). This article discusses the signs and symptoms of macroglossia, including the clinical and radiographic features, treatment techniques, as well as previously reported results. Cases are shown to illustrate the applicability of this technique. (Am J Orthod Dentofac Orthop 1996;110:170-7.)
An enlarged tongue (macroglossia) can cause dentomusculoskeletal deformities, instability of orthodontic and orthognathic surgical treatment, and create masticatory, speech, and airway management problems. Understanding the signs and symptoms of macroglossia will help identify those patients who could benefit from a reduction glossectomy (reduction of tongue size) to improve function, esthetics, and treatment stability. This article discusses the etiologic factors, diagnostic criteria, experimental studies, preferred technique for performing reduction glossectomy, and reported results. ETIOLOGIC FACTORS OF MACROGLOSSIA
Pseudomacroglossia is a condition where the tongue may be normal in size, but appears large relative to its anatomic interrelationships. This can be created by (1) habitual posturing of the tongue, (2) hypertrophied tonsils and adenoid tissue displacing the tongue forward, (3) low palatal vault decreasing the oral cavity volume, (4) transverse, vertical, or anteroposterior deficiency of the maxillary or mandibular arches that decreases the oral cavity volume, (5) severe mandibular deficiency, and (6) cysts or tumors that displace the tongue. Pseudomacroglossia must be distinguished from true macroglossia because the methods of manageaClinical Professor of Oral and MaxiUofacial Surgery, Baylor College of Dentistry; private Practice at Baylor University Medical Center. bFormer Fellow, Oral and Maxillofacial Surgery, Baylor College of Dentistry and Baylor University Medical Center; currently Assistant Professor of Oral and Maxillofaeial Surgery, Boston University Goldman School of Graduate Dentistry. Reprint requests to: Dr. Larry M. Wolford, 3409 Worth St., Suite 400, Dallas, TX 75246. Copyright © 1996 by the American Association of Orthodontists. 0889-5406/96/$5.00 + 0 $/1/60635
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ment may be different. For instance, if the problem is secondary to enlarged tonsils displacing the tongue forward, then the indicated treatment may be tonsillectomy, thereby increasing the oropharyngeal volume to accommodate the tongue. If the mandible is severely deficient, creating a relative macroglossia, then orthognathic surgery to advance the mandible would also increase the oral cavity volume. If a cyst or tumor is the etiologic factor, removal of the lesion would be indicated. There are many congenital and acquired causes of true macroglossia. Examples of congenital enlargement of the tongue include: (1) muscular hypertrophy, (2) glandular hyperplasia, (3) hemangioma, and (4) lymphangioma.1 Macroglossia also occurs commonly in conditions such as Downs Syndrome and Beckwith-Wiedemann syndrome. 2,3 Acquired factors may include acromegaly, myxedema, amyloidosis, tertiary syphilis, cyst or tumor involving the tongue, and neurologic injury.1"4With the acquired deformities, the underlying cause should be addressed primarily, and any residual macroglossia treated secondarily. By far, the most common cause of macroglossia is muscular hypertrophy and will be the primary focus of this article. Signs and Symptoms of Macroglossia
To determine whether a reduction glossectomy is necessary, it will be important to identify the signs and symptoms of macroglossia. There are several clinical and cephalometric features (Tables I and II) that may help the clinician identify the presence or absence of macroglossia. Not all of these features are always present and their exist-
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Table I, Clinical features Grossly enlarged and/or wide, broad and flat tongue Open bite (anterior or posterior) Mandibular prognathism Class III malocclusion with or without anterior and posterior crossbite Chronic posturing of the tongue between the teeth at rest (rule out habitual posturing of a normal sized tongue) Buccal tipping of posterior teeth (increased curve of Wilson in upper arch, reverse curve in lower arch) Accentuated curve of Spee in the maxillary arch Reverse curve of Spee in the mandibular arch Increased transverse width of mandibular and maxillary arches Diastemata in the mandibular or maxillary dentition Crenations on the tongue (scalloping) Glossitis (due to excessive mouth breathing) Speech articulation disorders Asymmetry in the maxillary or mandibular arches associated with an asymmetric tongue Difficulty eating and swallowing (severe cases) Instability in orthodontic mechanics or orthgnathic surgical procedures that in normal circumstances would be stable Airway difficulties, such as sleep apnea, secondary to oral or oropharyngeal obstruction Drooling
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-
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"
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-
Table Ih Cephalometric Radiographic features Tongue fills the oral cavity and extrudes through an anterior open bite Mandibular dentoalveolar protrusion or bimaxillary dentoalveolar protrusion Overangulation of the maxillary anterior teeth Overangulation of the mandibular anterior teeth Disproportionately excessive mandibular growth with dentoalveolar protrusion Decreased oropharyngeal airway Increased gonial angle Increased mandibular plane angle Increased mandibular occlusal plane angle
ence is not necessarily pathognomonic for the diagnosis of macroglossia. The tongue will reach its approximate adult size by the age of 8 years. 5 An evaluation of the tongue should include clinical, radiographic, and functional assessments relative to interference with speech, mastication, airway, and treatment stability. 4'6"8 Most open bites are not related to macroglossia. In fact, it has been established that closing open bites with orthognathic surgery will allow a normal tongue, which is a very adaptable organ, to readjust to the altered volume of the oral cavity with little tendency toward relapse. 6"9 If true macroglossia is present with the open bite, then instability of the orthodontics and orthognathic surgery may likely occur with a tendency for the open bite to return.
Fig. 1. Most common technique used for reduction glossectomy is "keyhole" or midline elliptical excision and anterior wedge resection. Incision edges are then sutured together in straight line.
Experimental Studies of the Tongue
Harvold wrote several articles relative to the role and function of enlargement or decreased size of the tongue. :° He artificially enlarged the tongue in growing primates by placing a palatal appliance with a large device on it to displace the tongue inferiorly and laterally, resulting in an anterior open bite and posterior crossbites. He also demonstrated that reducing the tongue to a size much smaller than normal causes the dental arches to collapse lingually. This important work shows the influences of the tongue on growth and development of the jaw and dentoosseous structures. Subtelny also demonstrated that enlarged tonsils can displace the tongue forward (pseudomacroglossia), creating an anterior open bite) 1 In pseudomacroglossia, tonsillectomy for removal of this etiologic factor may correct the open bite to some extent. Several articles have appeared describing various methods for reducing the tongue, including (1) the
172
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Fig. 2. A, Case 1. Lateral occlusion of patient with macroglossia is seen. Anterior open bite is present along with diastemas around upper lateral incisors and lower anterior teeth. B, At age 5 years 3 months (1 year 8 months after surgery) with no orthodontic intervention) open bite is seen to be decreased. C, Patient is seen at age of 7 years (3 years 5 months after surgery), showing essentially closure of open bite and closure of spacing. He has recently received cemented appliance in upper arch.
midline wedge resection with the base in the anterior tongue, (2) the midline elliptical excision, (3) the marginal excision, and (4) the "keyhole" or midline elliptical excision combined with an anterior wedge resection, have all been described. 12-18 The surgical technique that we most commonly used is the "keyhole" design (Fig. 1).
Fig. 3. A, Case 1. Patient is seen cephalometrically at age of 3 years 8 months. Note anterior open bite and Class III occlusal relationship. B, At age of 5 years 3 months (1 year 8 months after surgery), spontaneous closure of open bite is noted, with slight Class III occlusal tendency. C, At age of 7 years (3 years 5 months after surgery), he shows small open bite in occlusion and is still in mixed dentition.
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Fig. 4. A and B, Case 2. Woman, 31 years old, is seen with significant anterior open bite, but with good facial balance. C, She has significant macroglossia as observed with her mouth open. D and E, Patient is seen 18 months after treatment, with minimal change in facial esthetics.
Sequencing in Combination With Orthognathic Surgery In the presence of a musculoskeletal deformity with a malocclusion and true macroglossia, there are basically three choices on surgical sequencing: (1) Stage 1: reduction glossectomy, Stage 2: orthognathic surgery, (2) Stage 1: orthognathic surgery, Stage 2: reduction glossectomy and (3) perform the orthognathic surgery and reduction glossectomy in one surgical stage. The option of performing the reduction glossectomy first, as an isolated procedure, and the orthognathic surgery second has the following advantages as compared with a combined procedure: (1) less airway concern, (2) no intermaxillary fixation required, and (3) presurgical orthodontics, when performed after the reduction glossectomy, will be more stable and predictable. Relative indications for this sequencing can include patients with repeated functional distress (e.g., airway, mastication) and psychological concerns associated with the size of the tongue. An absolute indication is when extensive orthodontics are necessary before orthoguathic surgery, and the size of
the tongue impedes the required orthodontic movements. Reducing the size of the tongue in these cases is indicated to facilitate the stability of the presurgical orthodontics. The second sequencing option would be indicated if occlusal instability develops after orthodontics and orthognathic surgery. Development of dentoskeletal changes ,directly related with tongue size, such as an anterior open bite or a Class III occlusal tendency, would indicate that reduction glossectomy may be beneficial. In performing the treatment simultaneously, (option 3), with rigid fixation, it is usually helpful to complete the orthognathic surgery first. Once the orthognathic surgery is rigidly stabilized, a reduction glossectomy can then be performed. Because a reduction glossectomy generally causes a transient but significant increase in the size of the tongue, secondary to edema, performing the tongue procedure last may allow the occlusion to be better established before the onset of edema. However, if the tongue is extremely large, the reduction glossectomy may need to be sequenced first, to allow the proper occlu-
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Fig. 5. A and B, Preoperative occlusion is seen with anterior open bite extending from molar to molar. Lower right first molar is missing. Note diastemas, particularly in lower arch. C and D, Orthodontic setup aligned arches and collected all space in lower right side between second premolar and second molar. E and F, Postoperative stable occlusion observed, achieved by multiple maxillary osteotomies, bilateral mandibular sagittal osteotomies, right mandibular body ostectomy, and reduction glossectomy.
sion to be established when the orthognathic surgery is performed. The use of intermaxillary fixation for a few days will allow the teeth and jaws to act as a stent, thus, significantly decreasing the overall tongue edema. Also, when rigid fixation is used, if an airway problem does develop, the intermaxillary fixation can be removed to allow the patient to breathe more readily through the mouth. When using interosseous wiring, skeletal stabilization, and intermaxillary fixation techniques, the reduction glossectomy may best be performed at the beginning of surgery. Once the jaw structures are repositioned and stabilized, it can be difficult to perform the reduction glossectomy without displac-
ing some of the major jaw segments. The airway is a definite concern because the jaws are less stable, and therefore airway management is more critical and demanding. With rigid fixation, the ability to release the intermaxillary fixation, if necessary, is a significant advantage. There are potential risks and complications that can occur in reduction glossectomy including excessive bleeding, airway obstruction secondary to tongue edema, anesthesia of the tongue and loss of taste can develop secondary to lingual nerve injury, motor dysfunction secondary to hypoglossal nerve injury, decrease of tongue mobility secondary to scarring, salivary duct injury, and residual speech and masticatory problems.
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REPORTED RESULTS
With the glossectomy procedure described, mobility of the tongue will not be significantly decreased. 7 The lateral, downward, and protrusive movements will usually remain unchanged, although movement of the tongue cephalad may be somewhat restricted. The more muscle removed from the anterior tongue, the less upward mobility the tongue will retain. Large amounts of muscle can be removed from the central portion of the tongue without significantly restricting mobility. Stereognosis and oral motor ability after partial glossectomy has also been evaluated. 19 The surgery had a minor influence on the oral ability to recognize forms, with the incidence of false identifications increasing slightly. No decrease in oral motor ability was found. Taste sensation appears to be unaltered after glossectomy. 7 Even though the primary taste buds for sweetness are located in the anterior tongue, the other taste buds (sourness, bitterness, saltiness) seem to be stimulated sufficiently, by sweets, to provide the appropriate sensation. T h e r e are several published studies evaluating the results of reduction glossectomy procedures. Becker (22 cases), Kole (21 cases), Bjuggren (7 cases), Nordenram and Nordenram (16 cases) all reported good results with no complications. 1"12"2°-22 However, Egyedi and Obwegeser 13 reported on 18 cases with 14 patients who had decreased movement of the tongue, 7 patients who developed speech difficulties, and 2 patients with anesthesia of the tip of the tongue. In our experience, complications are very infrequent. CASE REPORTS Case 1
A 3-year, 8-month-old boy presented because of the development of an open bite and a worsening of his growth (observed by his parents), with his lower jaw becoming more prominent. He showed the signs and symptoms of an enlarged tongue, including anterior open bite, spacing between the teeth, and overangulation of lower incisors (Figs. 2, .4 and 3, A). The tongue, when relaxed, came out between his anterior teeth, and when the jaws were opened, the tongue covered his lower teeth almost totally. He also had some speech problems associated with the tongue size. A reduction glossectomy was performed and also a tonsillectomy. His occlusion, soon after, began closing together (Figs. 2, B and 3, B). At the age of 7 years (3 years and 5 months after surgery), his bite essentially closed, although he does have a slight Class III occlusal tendency yet (Figs. 2, C and 3, C). He
175
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Fig. 6. A, Case 2. Presurgical cephalometric tracing is seen. Note dotted line outlining position of tongue and also decreased oropharyngeal airway space of 5 mm (normal 11 mm + 2 mm). B, 18-month postsurgical cephalometric radiograph shows stability of results. Notice improved clearance between tongue and palate, as well as increase in oropharyngeal space. has recently began active orthodontic/orthopedic management. Case 2
This 31-year-old woman presented with difficulty eating and some mild speech problems. She also did not like the fact that her teeth did not fit together (Figs. 4, A and B, and 5,.4 and B). She had an accentuated curve of occlusion in the upper arch and a slight reversed curve of occlusion in the lower arch. She was missing the lower right first molar, with significant mesial drifting of the right second and third molars, and significant spacing between the teeth anteriorly, particularly on the right side of the mandibular arch (Fig. 5, A). The diagnosis
176
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American Journal of Orthodontics and Dentofacial Orthopedics August 1996
Fig. 7. A, B, and C, Case 3, This 16-year-old boy has Downs syndrome with mandibular prognathism and macroglossia. D, E, and F. Patient is seen 5 years after surgery, showing stability of mandibular setback and simultaneous reduction glossectomy.
consisted of the anterior open bite, slight mandibular prognathism, transverse maxillary deficiency, multiple spaces in the lower anterior arch, slight overangulation of lower incisors, and macroglossia (Figs. 4, C, and 6, A). The treatment included the following: (1) presurgical orthodontics (Fig. 5, B, C, and D), to align and level the maxillary arch and to align and level the mandibular arch, collecting all the edentulous space into the first molar area; (2) surgery: (a) multiple maxillary osteoto-
mies to expand it and to level the occlusal plane, (b) bilateral mandibular ramus osteotomies to rotate the mandible in an upward and forward direction, (c) right mandibular body osteotomy to remove the space in the first molar area and to move the second and third molars forward, and (d) reduction glossectomy; and (3) postsurgical orthodontics to finish and retain. The patient is seen 18 months after surgery, showing a good functional and occlusal result (Figs. 4, D and E, and 5, E and F, and 6, B).
Wolford and Cottrell 177
American Journal of Orthodontics and Dentofacial Orthopedics Volume 110, No. 2
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This 16-year-old boy was born with Downs Syndrome and had an associated mandibular prognathism, bilateral condylar hyperplasia, and macroglossia (Figs. 7, A, B, C, and 8, A). It was thought that the surgical procedure to correct his jaw deformity would be unstable if performed alone. Because of patient compliance difficulties, no orthodontics was used. The surgery included mandibular setback with bilateral ramus sagittal split osteotomies, with rigid fixation, bilateral high condylectomies to arrest the condylar hyperplastic growth, and reduction glossectomy. The patient is seen 5 years after surgery maintaining a good, stable result (Fig. 7, D, E, F, and 8, B). CONCLUSIONS
Although the indications for reduction glossectomy are few, when the procedure is indicated, the following conclusions can be appropriately drawn: 1. Reduction glossectomy can significantly improve functional and esthetic outcomes. 2. The anterior resection combined with the midline keyhole type procedure is the best technique. 3. Improved function relative to airway, speech, and mastication, can be anticipated. 4. If the excessively large tongue is causing significant, unfavorable mandibular growth, reduction of the tongue may help control the problem. REFERENCES
1. Bjuggren G, Jensen R, Strombeck JO. Macroglossia and its surgical treatment. Scand J Plast Reconstr 8urg I968;2:116-24.
2. McManamny D. Macroglossia as a presentation of the Beckwith-Wiedemann syndrome. Plast Reeonstr Surg I985;75:170-6. 3. Klaiman P, Witzel MA, Margar-Bacal F, Munro IR. Changes in aesthetic appearance and intelligibility of speech after partial glossectomy in patients with Down syndrome. Plast Reconstr Surg 1988;82(3):403-8. 4. Austermann KIt, Machtens E. The influence of tongue asymmetries on the development of jaws and the position of teeth. Int J Oral Surg 1974;3:261-5. 5. Proffit WR, Mason RM. Myofunctional therapy for tongue-thrusting: background and recommendations. J Am Dent Assoc 1975;90:403-I1. 6. Turvey TA, Joumot V, Epker BN. Correction of anterior open bite deformity: a study of tongue function, speech changes, and stability. J Maxillofac Snrg 1976;4:93-101. 7. Allison ML, Miller CW, Troiano MF, Wallace WR. Partial glossectomy for macroglossia. J Am Dent Asset 1971;82:852-7. 8. Fnjita S, Woodson BT, Clark JL, Wittig R. Laser midline glossectomy as a treatment for obstructive sleep apnea. Laryngoscope 1991;101:805-9. 9. Wickwire NA, White RP Jr, Proffit WR. The effect of mandibular osteotomy on tongue position. J Oral Surg I972;30:184-90. 10. Harvold EP. The role of function in the etiology and treatment of malocclusion. Am J Orthod I968;54:883-98. 1i. Subtelny JD, Sakunda M. Open-bite diagnosis and treatment. Am J Orthod I964;50:337-58. 12. Kole H. Results, experience, and problems in the operative treatment of anomalies with reverse overbite (mandibular protrusion). Oral Surg Oral Med Oral Pathol 1965;19:427-50. 13. Egyedi P, Obwegeser H. Znr operativen zungen-verldeinerung. Dtsch Zahn Mund Kieferheilk 1964;41:16-25. 14. Hendrick JW. Macrogiossia or giant tongue. Surgery 1956;39:674-7. 15. Rheinwald U. Die operative Znngenverkleinerung aus zahnartzlicher Indikation. Dtseh Zahn Mnnd Kieferheilk 1957;27:12%40. 16. Dingman R, Grabb W. Lymphangioma of the tongue. Plast Reconstr Surg 1961;27:214-23. 17. Egerton M. The management of macroglossia when associated with proguathism. Br J Plast Surg 1960;3:117-22. 18. Magee RB. Macroglossia. Am J Surg I962;103:632-5. i9. Ingervall B, Schmoker R. Effect of surgical reduction of the tongue on oral stereognosis, oral motor ability, and the rest position of the tongue and mandible. Am J Orthod Dentofac Orthop 1990;97:58-65. 20. Becker R. Ergebnisse bei der Behandlung der Progenie und des offenen Bisses bei gleichzeitiger Zungenverkleinerung. Dtsch Zahnarztl Zeitschr 1962;17:892903. 21. Nordenram A, Olow-Nordem'am M. Partial tongue excision in the treatment of apertognathia: part I. Oral Surg 1966;22:277-85. 22. Olow-Nordenram M, Nordenram A. Partial tongue excision in the treatment of apertognathia: part II. Oral Surg Oral Med Oral Pathol 1973;35:152-9.
Diagnosis of macroglossia and indications for reduction glossectomy Larry M. Wolford, DDS," and David A. Cottrell, DMD b Dallas, Texas, and Boston, Mass. Macroglossia can create dentomusculoskeletal deformities, instability of orthodontic and orthognathic surgical procedures, and masticatory, speech, and airway problems. The cause of macroglossia must be clearly defined, and true macroglossia separated from pseudomacroglossia (displacement of the tongue created by anatomic factors other than tongue size alone). This article discusses the signs and symptoms of macroglossia, including the clinical and radiographic features, treatment techniques, as well as previously reported results. Cases are shown to illustrate the applicability of this technique. (Am J Orthod Dentofac Orthop 1996;110:170-7.)
An enlarged tongue (macroglossia) can cause dentomusculoskeletal deformities, instability of orthodontic and orthognathic surgical treatment, and create masticatory, speech, and airway management problems. Understanding the signs and symptoms of macroglossia will help identify those patients who could benefit from a reduction glossectomy (reduction of tongue size) to improve function, esthetics, and treatment stability. This article discusses the etiologic factors, diagnostic criteria, experimental studies, preferred technique for performing reduction glossectomy, and reported results. ETIOLOGIC FACTORS OF MACROGLOSSIA
Pseudomacroglossia is a condition where the tongue may be normal in size, but appears large relative to its anatomic interrelationships. This can be created by (1) habitual posturing of the tongue, (2) hypertrophied tonsils and adenoid tissue displacing the tongue forward, (3) low palatal vault decreasing the oral cavity volume, (4) transverse, vertical, or anteroposterior deficiency of the maxillary or mandibular arches that decreases the oral cavity volume, (5) severe mandibular deficiency, and (6) cysts or tumors that displace the tongue. Pseudomacroglossia must be distinguished from true macroglossia because the methods of manageaClinical Professor of Oral and MaxiUofacial Surgery, Baylor College of Dentistry; private Practice at Baylor University Medical Center. bFormer Fellow, Oral and Maxillofacial Surgery, Baylor College of Dentistry and Baylor University Medical Center; currently Assistant Professor of Oral and Maxillofaeial Surgery, Boston University Goldman School of Graduate Dentistry. Reprint requests to: Dr. Larry M. Wolford, 3409 Worth St., Suite 400, Dallas, TX 75246. Copyright © 1996 by the American Association of Orthodontists. 0889-5406/96/$5.00 + 0 $/1/60635
170
ment may be different. For instance, if the problem is secondary to enlarged tonsils displacing the tongue forward, then the indicated treatment may be tonsillectomy, thereby increasing the oropharyngeal volume to accommodate the tongue. If the mandible is severely deficient, creating a relative macroglossia, then orthognathic surgery to advance the mandible would also increase the oral cavity volume. If a cyst or tumor is the etiologic factor, removal of the lesion would be indicated. There are many congenital and acquired causes of true macroglossia. Examples of congenital enlargement of the tongue include: (1) muscular hypertrophy, (2) glandular hyperplasia, (3) hemangioma, and (4) lymphangioma.1 Macroglossia also occurs commonly in conditions such as Downs Syndrome and Beckwith-Wiedemann syndrome. 2,3 Acquired factors may include acromegaly, myxedema, amyloidosis, tertiary syphilis, cyst or tumor involving the tongue, and neurologic injury.1"4With the acquired deformities, the underlying cause should be addressed primarily, and any residual macroglossia treated secondarily. By far, the most common cause of macroglossia is muscular hypertrophy and will be the primary focus of this article. Signs and Symptoms of Macroglossia
To determine whether a reduction glossectomy is necessary, it will be important to identify the signs and symptoms of macroglossia. There are several clinical and cephalometric features (Tables I and II) that may help the clinician identify the presence or absence of macroglossia. Not all of these features are always present and their exist-
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Table I, Clinical features Grossly enlarged and/or wide, broad and flat tongue Open bite (anterior or posterior) Mandibular prognathism Class III malocclusion with or without anterior and posterior crossbite Chronic posturing of the tongue between the teeth at rest (rule out habitual posturing of a normal sized tongue) Buccal tipping of posterior teeth (increased curve of Wilson in upper arch, reverse curve in lower arch) Accentuated curve of Spee in the maxillary arch Reverse curve of Spee in the mandibular arch Increased transverse width of mandibular and maxillary arches Diastemata in the mandibular or maxillary dentition Crenations on the tongue (scalloping) Glossitis (due to excessive mouth breathing) Speech articulation disorders Asymmetry in the maxillary or mandibular arches associated with an asymmetric tongue Difficulty eating and swallowing (severe cases) Instability in orthodontic mechanics or orthgnathic surgical procedures that in normal circumstances would be stable Airway difficulties, such as sleep apnea, secondary to oral or oropharyngeal obstruction Drooling
,/ V
,\
-
2
,?
ix?
a
"
;:
-
Table Ih Cephalometric Radiographic features Tongue fills the oral cavity and extrudes through an anterior open bite Mandibular dentoalveolar protrusion or bimaxillary dentoalveolar protrusion Overangulation of the maxillary anterior teeth Overangulation of the mandibular anterior teeth Disproportionately excessive mandibular growth with dentoalveolar protrusion Decreased oropharyngeal airway Increased gonial angle Increased mandibular plane angle Increased mandibular occlusal plane angle
ence is not necessarily pathognomonic for the diagnosis of macroglossia. The tongue will reach its approximate adult size by the age of 8 years. 5 An evaluation of the tongue should include clinical, radiographic, and functional assessments relative to interference with speech, mastication, airway, and treatment stability. 4'6"8 Most open bites are not related to macroglossia. In fact, it has been established that closing open bites with orthognathic surgery will allow a normal tongue, which is a very adaptable organ, to readjust to the altered volume of the oral cavity with little tendency toward relapse. 6"9 If true macroglossia is present with the open bite, then instability of the orthodontics and orthognathic surgery may likely occur with a tendency for the open bite to return.
Fig. 1. Most common technique used for reduction glossectomy is "keyhole" or midline elliptical excision and anterior wedge resection. Incision edges are then sutured together in straight line.
Experimental Studies of the Tongue
Harvold wrote several articles relative to the role and function of enlargement or decreased size of the tongue. :° He artificially enlarged the tongue in growing primates by placing a palatal appliance with a large device on it to displace the tongue inferiorly and laterally, resulting in an anterior open bite and posterior crossbites. He also demonstrated that reducing the tongue to a size much smaller than normal causes the dental arches to collapse lingually. This important work shows the influences of the tongue on growth and development of the jaw and dentoosseous structures. Subtelny also demonstrated that enlarged tonsils can displace the tongue forward (pseudomacroglossia), creating an anterior open bite) 1 In pseudomacroglossia, tonsillectomy for removal of this etiologic factor may correct the open bite to some extent. Several articles have appeared describing various methods for reducing the tongue, including (1) the
172
Wolford and Cottrell
American Journal of Orthodontics and Dentofacial Orthopedics August 1996
BLG Age: 3.8 yrs
t_
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A BLG Age: 5.3 yrs
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Fig. 2. A, Case 1. Lateral occlusion of patient with macroglossia is seen. Anterior open bite is present along with diastemas around upper lateral incisors and lower anterior teeth. B, At age 5 years 3 months (1 year 8 months after surgery) with no orthodontic intervention) open bite is seen to be decreased. C, Patient is seen at age of 7 years (3 years 5 months after surgery), showing essentially closure of open bite and closure of spacing. He has recently received cemented appliance in upper arch.
midline wedge resection with the base in the anterior tongue, (2) the midline elliptical excision, (3) the marginal excision, and (4) the "keyhole" or midline elliptical excision combined with an anterior wedge resection, have all been described. 12-18 The surgical technique that we most commonly used is the "keyhole" design (Fig. 1).
Fig. 3. A, Case 1. Patient is seen cephalometrically at age of 3 years 8 months. Note anterior open bite and Class III occlusal relationship. B, At age of 5 years 3 months (1 year 8 months after surgery), spontaneous closure of open bite is noted, with slight Class III occlusal tendency. C, At age of 7 years (3 years 5 months after surgery), he shows small open bite in occlusion and is still in mixed dentition.
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Fig. 4. A and B, Case 2. Woman, 31 years old, is seen with significant anterior open bite, but with good facial balance. C, She has significant macroglossia as observed with her mouth open. D and E, Patient is seen 18 months after treatment, with minimal change in facial esthetics.
Sequencing in Combination With Orthognathic Surgery In the presence of a musculoskeletal deformity with a malocclusion and true macroglossia, there are basically three choices on surgical sequencing: (1) Stage 1: reduction glossectomy, Stage 2: orthognathic surgery, (2) Stage 1: orthognathic surgery, Stage 2: reduction glossectomy and (3) perform the orthognathic surgery and reduction glossectomy in one surgical stage. The option of performing the reduction glossectomy first, as an isolated procedure, and the orthognathic surgery second has the following advantages as compared with a combined procedure: (1) less airway concern, (2) no intermaxillary fixation required, and (3) presurgical orthodontics, when performed after the reduction glossectomy, will be more stable and predictable. Relative indications for this sequencing can include patients with repeated functional distress (e.g., airway, mastication) and psychological concerns associated with the size of the tongue. An absolute indication is when extensive orthodontics are necessary before orthoguathic surgery, and the size of
the tongue impedes the required orthodontic movements. Reducing the size of the tongue in these cases is indicated to facilitate the stability of the presurgical orthodontics. The second sequencing option would be indicated if occlusal instability develops after orthodontics and orthognathic surgery. Development of dentoskeletal changes ,directly related with tongue size, such as an anterior open bite or a Class III occlusal tendency, would indicate that reduction glossectomy may be beneficial. In performing the treatment simultaneously, (option 3), with rigid fixation, it is usually helpful to complete the orthognathic surgery first. Once the orthognathic surgery is rigidly stabilized, a reduction glossectomy can then be performed. Because a reduction glossectomy generally causes a transient but significant increase in the size of the tongue, secondary to edema, performing the tongue procedure last may allow the occlusion to be better established before the onset of edema. However, if the tongue is extremely large, the reduction glossectomy may need to be sequenced first, to allow the proper occlu-
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American Journal of Orthodontics and Dentofacial Orthopedics August 1996
Fig. 5. A and B, Preoperative occlusion is seen with anterior open bite extending from molar to molar. Lower right first molar is missing. Note diastemas, particularly in lower arch. C and D, Orthodontic setup aligned arches and collected all space in lower right side between second premolar and second molar. E and F, Postoperative stable occlusion observed, achieved by multiple maxillary osteotomies, bilateral mandibular sagittal osteotomies, right mandibular body ostectomy, and reduction glossectomy.
sion to be established when the orthognathic surgery is performed. The use of intermaxillary fixation for a few days will allow the teeth and jaws to act as a stent, thus, significantly decreasing the overall tongue edema. Also, when rigid fixation is used, if an airway problem does develop, the intermaxillary fixation can be removed to allow the patient to breathe more readily through the mouth. When using interosseous wiring, skeletal stabilization, and intermaxillary fixation techniques, the reduction glossectomy may best be performed at the beginning of surgery. Once the jaw structures are repositioned and stabilized, it can be difficult to perform the reduction glossectomy without displac-
ing some of the major jaw segments. The airway is a definite concern because the jaws are less stable, and therefore airway management is more critical and demanding. With rigid fixation, the ability to release the intermaxillary fixation, if necessary, is a significant advantage. There are potential risks and complications that can occur in reduction glossectomy including excessive bleeding, airway obstruction secondary to tongue edema, anesthesia of the tongue and loss of taste can develop secondary to lingual nerve injury, motor dysfunction secondary to hypoglossal nerve injury, decrease of tongue mobility secondary to scarring, salivary duct injury, and residual speech and masticatory problems.
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REPORTED RESULTS
With the glossectomy procedure described, mobility of the tongue will not be significantly decreased. 7 The lateral, downward, and protrusive movements will usually remain unchanged, although movement of the tongue cephalad may be somewhat restricted. The more muscle removed from the anterior tongue, the less upward mobility the tongue will retain. Large amounts of muscle can be removed from the central portion of the tongue without significantly restricting mobility. Stereognosis and oral motor ability after partial glossectomy has also been evaluated. 19 The surgery had a minor influence on the oral ability to recognize forms, with the incidence of false identifications increasing slightly. No decrease in oral motor ability was found. Taste sensation appears to be unaltered after glossectomy. 7 Even though the primary taste buds for sweetness are located in the anterior tongue, the other taste buds (sourness, bitterness, saltiness) seem to be stimulated sufficiently, by sweets, to provide the appropriate sensation. T h e r e are several published studies evaluating the results of reduction glossectomy procedures. Becker (22 cases), Kole (21 cases), Bjuggren (7 cases), Nordenram and Nordenram (16 cases) all reported good results with no complications. 1"12"2°-22 However, Egyedi and Obwegeser 13 reported on 18 cases with 14 patients who had decreased movement of the tongue, 7 patients who developed speech difficulties, and 2 patients with anesthesia of the tip of the tongue. In our experience, complications are very infrequent. CASE REPORTS Case 1
A 3-year, 8-month-old boy presented because of the development of an open bite and a worsening of his growth (observed by his parents), with his lower jaw becoming more prominent. He showed the signs and symptoms of an enlarged tongue, including anterior open bite, spacing between the teeth, and overangulation of lower incisors (Figs. 2, .4 and 3, A). The tongue, when relaxed, came out between his anterior teeth, and when the jaws were opened, the tongue covered his lower teeth almost totally. He also had some speech problems associated with the tongue size. A reduction glossectomy was performed and also a tonsillectomy. His occlusion, soon after, began closing together (Figs. 2, B and 3, B). At the age of 7 years (3 years and 5 months after surgery), his bite essentially closed, although he does have a slight Class III occlusal tendency yet (Figs. 2, C and 3, C). He
175
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Fig. 6. A, Case 2. Presurgical cephalometric tracing is seen. Note dotted line outlining position of tongue and also decreased oropharyngeal airway space of 5 mm (normal 11 mm + 2 mm). B, 18-month postsurgical cephalometric radiograph shows stability of results. Notice improved clearance between tongue and palate, as well as increase in oropharyngeal space. has recently began active orthodontic/orthopedic management. Case 2
This 31-year-old woman presented with difficulty eating and some mild speech problems. She also did not like the fact that her teeth did not fit together (Figs. 4, A and B, and 5,.4 and B). She had an accentuated curve of occlusion in the upper arch and a slight reversed curve of occlusion in the lower arch. She was missing the lower right first molar, with significant mesial drifting of the right second and third molars, and significant spacing between the teeth anteriorly, particularly on the right side of the mandibular arch (Fig. 5, A). The diagnosis
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American Journal of Orthodontics and Dentofacial Orthopedics August 1996
Fig. 7. A, B, and C, Case 3, This 16-year-old boy has Downs syndrome with mandibular prognathism and macroglossia. D, E, and F. Patient is seen 5 years after surgery, showing stability of mandibular setback and simultaneous reduction glossectomy.
consisted of the anterior open bite, slight mandibular prognathism, transverse maxillary deficiency, multiple spaces in the lower anterior arch, slight overangulation of lower incisors, and macroglossia (Figs. 4, C, and 6, A). The treatment included the following: (1) presurgical orthodontics (Fig. 5, B, C, and D), to align and level the maxillary arch and to align and level the mandibular arch, collecting all the edentulous space into the first molar area; (2) surgery: (a) multiple maxillary osteoto-
mies to expand it and to level the occlusal plane, (b) bilateral mandibular ramus osteotomies to rotate the mandible in an upward and forward direction, (c) right mandibular body osteotomy to remove the space in the first molar area and to move the second and third molars forward, and (d) reduction glossectomy; and (3) postsurgical orthodontics to finish and retain. The patient is seen 18 months after surgery, showing a good functional and occlusal result (Figs. 4, D and E, and 5, E and F, and 6, B).
Wolford and Cottrell 177
American Journal of Orthodontics and Dentofacial Orthopedics Volume 110, No. 2
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Age: 14.2 yrs Pre treatment
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Fig. 8. A, Case 3. Pretreatment cephalometr.'~, analysis shows prognathie appearance of mandible. B, Five-year follow-up cephalometric radiograpl% shows stability of results of combined procedures. Case 3
This 16-year-old boy was born with Downs Syndrome and had an associated mandibular prognathism, bilateral condylar hyperplasia, and macroglossia (Figs. 7, A, B, C, and 8, A). It was thought that the surgical procedure to correct his jaw deformity would be unstable if performed alone. Because of patient compliance difficulties, no orthodontics was used. The surgery included mandibular setback with bilateral ramus sagittal split osteotomies, with rigid fixation, bilateral high condylectomies to arrest the condylar hyperplastic growth, and reduction glossectomy. The patient is seen 5 years after surgery maintaining a good, stable result (Fig. 7, D, E, F, and 8, B). CONCLUSIONS
Although the indications for reduction glossectomy are few, when the procedure is indicated, the following conclusions can be appropriately drawn: 1. Reduction glossectomy can significantly improve functional and esthetic outcomes. 2. The anterior resection combined with the midline keyhole type procedure is the best technique. 3. Improved function relative to airway, speech, and mastication, can be anticipated. 4. If the excessively large tongue is causing significant, unfavorable mandibular growth, reduction of the tongue may help control the problem. REFERENCES
1. Bjuggren G, Jensen R, Strombeck JO. Macroglossia and its surgical treatment. Scand J Plast Reconstr 8urg I968;2:116-24.
2. McManamny D. Macroglossia as a presentation of the Beckwith-Wiedemann syndrome. Plast Reeonstr Surg I985;75:170-6. 3. Klaiman P, Witzel MA, Margar-Bacal F, Munro IR. Changes in aesthetic appearance and intelligibility of speech after partial glossectomy in patients with Down syndrome. Plast Reconstr Surg 1988;82(3):403-8. 4. Austermann KIt, Machtens E. The influence of tongue asymmetries on the development of jaws and the position of teeth. Int J Oral Surg 1974;3:261-5. 5. Proffit WR, Mason RM. Myofunctional therapy for tongue-thrusting: background and recommendations. J Am Dent Assoc 1975;90:403-I1. 6. Turvey TA, Joumot V, Epker BN. Correction of anterior open bite deformity: a study of tongue function, speech changes, and stability. J Maxillofac Snrg 1976;4:93-101. 7. Allison ML, Miller CW, Troiano MF, Wallace WR. Partial glossectomy for macroglossia. J Am Dent Asset 1971;82:852-7. 8. Fnjita S, Woodson BT, Clark JL, Wittig R. Laser midline glossectomy as a treatment for obstructive sleep apnea. Laryngoscope 1991;101:805-9. 9. Wickwire NA, White RP Jr, Proffit WR. The effect of mandibular osteotomy on tongue position. J Oral Surg I972;30:184-90. 10. Harvold EP. The role of function in the etiology and treatment of malocclusion. Am J Orthod I968;54:883-98. 1i. Subtelny JD, Sakunda M. Open-bite diagnosis and treatment. Am J Orthod I964;50:337-58. 12. Kole H. Results, experience, and problems in the operative treatment of anomalies with reverse overbite (mandibular protrusion). Oral Surg Oral Med Oral Pathol 1965;19:427-50. 13. Egyedi P, Obwegeser H. Znr operativen zungen-verldeinerung. Dtsch Zahn Mund Kieferheilk 1964;41:16-25. 14. Hendrick JW. Macrogiossia or giant tongue. Surgery 1956;39:674-7. 15. Rheinwald U. Die operative Znngenverkleinerung aus zahnartzlicher Indikation. Dtseh Zahn Mnnd Kieferheilk 1957;27:12%40. 16. Dingman R, Grabb W. Lymphangioma of the tongue. Plast Reconstr Surg 1961;27:214-23. 17. Egerton M. The management of macroglossia when associated with proguathism. Br J Plast Surg 1960;3:117-22. 18. Magee RB. Macroglossia. Am J Surg I962;103:632-5. i9. Ingervall B, Schmoker R. Effect of surgical reduction of the tongue on oral stereognosis, oral motor ability, and the rest position of the tongue and mandible. Am J Orthod Dentofac Orthop 1990;97:58-65. 20. Becker R. Ergebnisse bei der Behandlung der Progenie und des offenen Bisses bei gleichzeitiger Zungenverkleinerung. Dtsch Zahnarztl Zeitschr 1962;17:892903. 21. Nordenram A, Olow-Nordem'am M. Partial tongue excision in the treatment of apertognathia: part I. Oral Surg 1966;22:277-85. 22. Olow-Nordenram M, Nordenram A. Partial tongue excision in the treatment of apertognathia: part II. Oral Surg Oral Med Oral Pathol 1973;35:152-9.