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Vol. 95 No. 3 March 2003
ORAL SURGERY
ORAL MEDICINE
ORAL PATHOLOGY
REVIEW ARTICLE
The role of tongue reduction
Joy Wang, DMD, MD, MPH,a Nicholas M. Goodger, FRCS, FDS, DLORCS,a and
M. Anthony Pogrel, DDS, MD,b San Francisco, Calif
UNIVERSITY OF CALIFORNIA, SAN FRANCISCO
Macroglossia may occur as a congenital or acquired condition. The enlarged tongue has both functional and
cosmetic deformity, which may affect the oral airway, speech, and the development of the jaws. We discuss the
various tongue-reduction procedures and present cases to illustrate the uses of this procedure, its potential
complications, and the results. (Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2003;95:269-73)
Large tongue, or macroglossia, is an uncommon congenital condition caused by vascular malformation,
congenital syndromes such as Beckwith-Wiedemann,
and muscular hypertrophy.1-13 It is also found as a rare
acquired state in conditions such as amyloidosis, neurofibromatosis, acromegaly, hypothyroidism, and chronic
edema.1,5,14-18 Surgical reduction of the tongue is only
occasionally required, but it may be indicated in cases of
congenital macroglossia,1-5,8 Beckwith-Wiedemann syndrome,9-13 and before or after orthodontic treatment or
orthognathic surgery to prevent relapse caused by an enlarged or dysfunctional tongue,19,20 the macroglossia of
amyloid,14-16 or chronic edema. We describe the surgical
techniques, complications, and outcomes.
Many surgical incisions have been proposed to accomplish tongue reduction (Fig 1). Peripheral excisions
such as those described by Butlin5 and Ensign15 (Fig 1,
A) or by Harris21 and Dingman and Grabb2 (Fig 1, D)
leave the tongue globular and immobile. A V-shaped
wedge taken from the tip of the tongue, as reported by
Harris,22 Blair,23 and Hendrick3 (Fig 1, B), will shorten
but not narrow it, whereas an ellipse taken from the
midline of the tongue will narrow it but not shorten it
(Fig 1, C).24 Pichler and Trauner25 proposed a combia
Resident, Department of Oral and Maxillofacial Surgery, University
of California, San Francisco.
b
Professor and Chairman, Department of Oral and Maxillofacial
Surgery, University of California, San Francisco.
Received for publication May 8, 2002; returned for revision Jul 10,
2002; accepted for publication Sep 16, 2002.
© 2003, Mosby, Inc.
1079-2104/2003/$30.00 ⫹ 0
doi:10.1067/moe.2003.1
nation of an ellipse from the midline posteriorly combined with a wedge from the tip, and various other
incisions have been recorded,10,26-31 but the so-called
keyhole excision allows both narrowing and shortening
of the tongue as required (Fig 1, L).32 Care should be
taken to avoid the lingual arteries, but bleeding may be
brisk despite this. Even if both lingual arteries are
severed, the tongue will not normally necrose because
of the rich collateral blood supply and anastomoses.
Postoperative edema is often quite substantial, and sutures should not be too small or too tight because the
swelling may cause them to cut through. This particular
technique is illustrated by the 3 following cases.
CASE REPORTS
Case 1
A 32-year-old man presented with a 3-year history of
massive lymphedema of the tongue after acute respiratory
distress syndrome and renal failure secondary to necrotic
pancreatitis. The macroglossia and associated right-sided 12th
nerve palsy (caused by compression injury from the edema)
resulted in a speech impediment and marked proclination of
the anterior teeth, in addition to potential airway embarrassment (Fig 2). A tongue-reduction procedure was requested before the commencement of orthodontic treatment. A large keyhole-type excision resulted in a great improvement in tongue size
and allowed orthodontic treatment to proceed. Despite the good
result from initial surgery (Fig 3) and orthodontic correction of
the maxillary teeth, orthognathic surgery was required to close
the residual anterior open bite. A further tongue-reduction procedure was required; again the keyhole excision was used. This
was followed by a Le Fort I osteotomy to correct the residual
anterior open bite without relapse (Fig 4).
269
270 Wang, Goodger, and Pogrel
ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGY
March 2003
Fig 1. Tongue-reduction procedures. A, Butlin5 and Ensign15; B, Harris,22 Blair,23 and Hendrick3; C, Edgerton24; D, Dingman and
Grabb2 and Gupta8; E, Egyedi and Obwegeser26; F, Köle27 and Davalbhakta and Lamberty10; G and H, Austerman and
Machtens28; I, Kruchinsky29; J, Mixter et al30; K, Harada and Enomoto31; L, Morgan et al32 and Kacker et al.9
Case 2
Beckwith-Wiedemann syndrome (BWS) is characterized
by macroglossia, umbilical hernia, exomphalos, nephromegaly, and Leydig cell hyperplasia.9,11,12 The prevalence of
BWS has been estimated to be 0.07 per 1000 births. However,
it is felt that this number is underestimated because of the
early death of children who have yet to manifest the signs of
BWS or because less-affected patients may not have been
classified.9 Patients with BWS come to the attention of the
oral and maxillofacial surgeon for the treatment of upper
airway obstruction secondary to macroglossia. There are several temporary treatment options for airway management of
these children, such as the use of oral splints or laryngeal
mask airways. The definitive treatment usually consists of
partial glossectomy with or without tracheostomy. Respiratory distress in these patients may be complicated by concurrent abdominal masses such as hepatoblastoma or insulinoma
that can result in decreased total lung capacity and tidal
volume.
A 14-week-old male infant (34-week gestational age) whose
amniocentesis had shown a normal karyotype was diagnosed
with BWS after transfer from a Northern California hospital.
Before transfer he was dehydrated and not thriving, and he had
persistent hypoglycemia. On admission he was found to have
macroglossia (Fig 5) and increased abdominal girth secondary to
hemorrhage from a hepatoblastoma.
Five days after admission, the infant had increased respiratory distress requiring surgical intervention. He underwent
ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGY
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Fig 2. Patient 1. Macroglossia and proclination of the lower
anterior teeth were seen at presentation.
Wang, Goodger, and Pogrel 271
Fig 4. Patient 1. Final occlusion.
Fig 3. Patient 1. Postoperative appearance of the tongue after
initial tongue reduction.
Fig 5. Patient 2. Preoperative appearance of macroglossia.
a tracheostomy and a keyhole tongue reduction. Despite the
friable nature of the tissue, which made approximation difficult, the tongue was closed with several layers. After surgery,
the patient was sedated. Perioperative and postoperative
methylprednisolone was given to reduce surgical edema. The
tongue was well healed at 6 weeks, and the patient was able
to keep his tongue in his oral cavity (Fig 6).
Case 3
An 18-year-old girl with developmental delay, facial dysmorphism, and polycystic ovarian disease was referred for
craniofacial assessment of an anterior open bite and generalized anterior spacing. A tongue guard had been placed 3
months before presentation to alter tongue function; although
this allowed closure of the anterior open bite, a bilateral
posterior open bite resulted. It was felt that the tongue posture
and size had caused this change. After the removal of the
tongue guard, the posterior open bite closed, but the anterior
open bite recurred. To secure a stable orthodontic environment, a keyhole-type reduction glossectomy was performed
with the patient under general anesthesia (Figs 7-9), which
allowed her orthodontic treatment to proceed without relapse.
DISCUSSION
Macroglossia was first described by Galen in the second century21 and is illustrated in medieval carvings.34 A
number of cases are also recorded through the 16th and
17th centuries.21,22 The first report of congenital macroglossia secondary to a lymphatic hamartoma was in
1854.4 Surgical treatment of macroglossia caused by mercury poisoning was reported as early as 1658,33 and Bar-
272 Wang, Goodger, and Pogrel
ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGY
March 2003
Fig 8. Patient 3. Surgical specimen.
Fig 6. Patient 2. Postoperative appearance of the tongue at 6
weeks.
Fig 9. Patient 3. Immediate postoperative appearance.
Fig 7. Patient 3. Tongue incision is marked.
tholin operated on a patient with macroglossia in 1680.22
Early surgical techniques (illustrated in Fig 1, A-D) were
aimed at the reduction of tongue bulk. Those procedures
described more recently were aimed at the preservation of
tongue function, along with the reduction of tongue mass.
The incisions shown in Fig 1, I-K, were designed to
preserve the tip of the tongue.29-31 The keyhole procedure
allows for greater reduction of the tongue mass, and the
loss of the tip is not reported to result in a marked sensory
deficit, which is confirmed in our experience.9,32 Other
procedures have been proposed: Davalbhakta and Lamberty10 reported a modification of the Köle incision (Fig 1,
F) with horizontal wedges taken from the muscle laterally
to ensure the reduction of tongue thickness, and Austerman and Machtens10 described 2 methods for the reduction of asymmetric tongue enlargement (Fig 1, G and
H).28 With central tongue-reduction procedures, such as
the keyhole excision, the resection must extend to just
anterior of the circumvallate papillae for adequate reduction, but particular care must be taken to avoid the neurovascular bundles, which run inferolaterally.35
Taste and tongue mobility are rarely affected by tongue
reduction.10,20 Speech can be affected, but this complication usually corrects itself within a matter of weeks and
formal speech therapy is rarely needed. Speech improvement has been reported as a result of tongue reduction.36
Sounds made primarily by the tip of the tongue against the
upper or lower teeth or the anterior palate are affected.37
Wang, Goodger, and Pogrel 273
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The sensitivity of the tongue tip is also sometimes altered,
but, again, this usually resolves spontaneously within a
few weeks. Revision surgery may be required if wound
breakdown and subsequent secondary healing caused by
postoperative edema cause either a bifid tongue tip or
clefting of the tongue.
The indications for tongue reduction are relatively few.
In connection with orthodontics and orthognathic surgery,
most authorities recommend proceeding with the treatment and carry out a tongue reduction after treatment only
if the tongue appears to be instrumental in causing relapse.19 In these cases, the tongue reduction is carried out
at the first sign of relapse but is actually required relatively
infrequently. However, in our first patient, the tongue
enlargement was so extreme that orthodontics could not
be commenced until the tongue was reduced. Several
articles illustrate the spontaneous correction of anterior
open bite after tongue reduction.28,34,38 In BWS, tongue
reduction can allow decannulation and more normal development,9 and in other conditions such as amyloidosis,
it may improve both function and cosmesis.
CONCLUSION
Tongue reduction is an uncommon procedure that may
be required for functional and esthetic reasons. The aim of
this surgery is to produce a tongue that lies within the
lower dental arch and yet can protrude to wet the lips. In
our experience, the keyhole excision provides a flexible
template to narrow or shorten the tongue as required.
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Reprint requests:
M. A. Pogrel, DDS, MD
Box 0440
521 Parnassus Ave
San Francisco, CA 94143-0440
[email protected]