Tongue lesions in children

Tongue Lesions in Children By Francisca T. Velcek, Donald H. Klotz, Constance H. Hill, Loredana E. Ladogana, and Peter K. Kottmeier Brooklyn, New York 9 A review of tongue lesions in children showed that there is a great variety requiring operative intervention, often in infancy. While the presenting symptoms may be related to dysphagia and dyspnea, the aim of operative intervention should not only be to salvage life by restoration of breathing and swallowing, but also to leave a tongue capable of adequate speech, taste, sensation, and normal orofacial development. Intimate knowledge of lingual anatomy and function is necessary to allow selection of the ideal procedure and appropriate timing of the therapy. While careful observation and nonoperative approach may be indicated in non-neoplastic macroglossia, early intervention is often necessary in diffuse neoplastic lesions such as lymphangioma, fibromatosis, or fibrolipomatous dysplasia. While malignant tumors are rare in childhood, they do occur and have to be ruled out. INDEX WORDS: Tongue lesions; tongue tumors.

AGE OF surgical specialization, only I Na THE few anatomic areas have remained unclaimed. The tongue is one of these areas and few comprehensive reports on lingual lesions in childhood exist. A review of children with lingual problems seen by our Pediatric Surgical Service showed that the wide variety of tongue lesions occurring in children demands individual evaluation leading to different diagnostic and therapeutic approaches for lesions that at first glance might appear to be similar. We, therefore, reviewed all children operated upon for tongue lesions on our service and the experience of others to correlate the various types of tongue lesions in childhood with age, symptomatology, diagnosis and therapy and, in particular, the From the Pediatric Surgical Division, Department of Surgery, and The Department of Anesthesiology, State University of New York Downstate Medical Center, Brooklyn, N.Y. 11203. Presented before the 27th Annual Meeting of the Surgical Section of the American Academy of Pediatrics, Chicago, Hlinois, October 22-23, 1978. Address reprint requests to Peter K. Kottmeier, M.D., Professor and Chief Pediatric Surgery, State University of New York, Downstate Medical Center, 450 Clarkson Avenue, Brooklyn, N.Y. 11203. 9 1979 by Grune & Stratton, Inc. 0022-3468/79/1403-0005501.00/(9 238

relationship between operative technique and postoperative function. MATERIALS AND METHODS As shown in Table I, 18 children were operated upon to correct a variety of congenital, neoplastic or acquired lingual lesions.

Symptoms Severe respiratory symptoms occurred in all infants born with lymphangiomas involving the tongue. In all of these infants, the lymphangioma not only involved the tongue alone, but also the floor of the mouth and/or neck to varying degrees. All of these infants required tracheostomies, all but one after oral-~or nasal--intubation. Moderate respiratory difficulties were present in a child with a lingual thyroid (Patient 2) and in two patients with lingual cysts (Patients 3 and 4). The more slowly growing solid lesions (Patients 9-13) did not present with significant respiratory difficulties. Dysphagia was present in almost all children, regardless of the underlying etiology. Most of the children were unable to swallow solid food at the time of admission. The degree of dysphagia was usually proportional to the extent of the patient's dyspnea and most pronounced in infants with lymphangiomas. Salivation or "drooling", however, was not directly related to the extent of the lingual tumor or trauma. Marked salivation was occasionally present in children with relatively minor lesions. Since 9 of the lesions requiring operative intervention occurred in children under the age of 2 yr, preoperative speech evaluation is, therefore, difficult. In children with well-delineated lesions, there was little interference with speech. Whatever speech difficulty was present, it appeared to be related to the site of the lesion rather than the size. Extensive lesions, such as the fibrolipomatous dysplasia in Patient 13, led to marked speech interference.

Anesthesia All children were anesthetized prior to intubation or tracheostomy. With the exception of one infant with an extensive head and neck lymphangioma, where endotracheal intubation was impossible, all children underwent an oral or nasotracheal intubation, followed by tracheostomy in six patients, lntubation was particularly difficult in children with lymphangiomas, where the involvement of the floor of the mouth led to an elevation of the tongue, obliterating the view of larynx and vocal cords. Tracheostomies in children where the neck was diffusely infiltrated by the lymphangioma were equally difficult and should, therefore, only be performed after intubation, if at all possible. In contrast to the children with diffuse lymphangiomas, the other congenital or acquired lesions did not present a major airway problem, even after extensive glossorrhaphies.

Journal of Pediatric Surgery, Vol. 14, No. 3 (June), 1979

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Table 1. Tongue Lesions Type Congenital Cystic

Solid

Trauma

Diagnosis Macroglossia Lingual thyroid Solitary lingual cyst Lingual cyst Dermoid (dumbbell) Lymphangioma Lymphangioma Lyrnphangioma Lymphangioma Rhabdomyoma Neurofibroma Fibromatosis Granular cell myoblastoma Fibrolipomatous dysplasia Laceration Laceration Laceration Laceration Laceration

Patient Number

Age

Sex

Tracheotomy

1 2 3 4

9m 10 (16) 5 2m

F F M M

5 6 7 8 9 10 11 12

NB NB NB NB 11 8 4m 4d

M F M M M F F F

No Yes No No No Yes Yes Yes Yes No No Yes No

Fillet Pedicle transplant Excision Excision Excision Fillet (staged) Fillet (staged) Sublingual excision Fillet Glossoplasty Glossoplasty Glossoplasty Glossoplasty

A, M P A, M, P A, M A, Sublingual A, M, Neck A, M, Neck A, M, P, Neck A, M, Neck A M, P A, M A, M

13

10

M

Yes

Glossoplasty

A, M

14 15 16 17 18

4 8 6 42/z 1 ~/2

M M F M M

No No No No No

Glossorrhaphy Glossorrhaphy Glossorrhaphy GIossorrhaphy Glossorrhaphy, cheiloplasty

Treatment

One-Third of Tongue*

*A -- Anterior, M = Medial, P = Posterior third of tongue.

Although lingual edema was quite pronounced after surgical intervention, it rarely led to major respiratory difficulties, since in the majority of cases the operative intervention was limited to the anterior and mid-third of the tongue. The posloperative edema was therefore limited, leaving the nasal airway open. The edema subsided in almost all patients within 48 hr and endotracheal intubation was discontinued in most children within 24-48 hr.

DISCUSSION

No other muscular part of the body provides such a unique and complicated interaction between various muscles and the peripheral and central nervous system, as the tongue, involving speech, taste, sensation, mastication, and respiration. Intimate knowledge of the functional components of the tongue is, therefore, necessary to correlate treatment, pathology, and pre- and postoperative function.

Anatomy The muscular components of the tongue consist of two major groups. The extrinsic group is responsible for the gross position of the body of the tongue (genioglossus, hyoglossus, styloglossus, and palatoglossus). The intrinsic group determines the shape of the tongue (superior and inferior longitudinal muscles, transverse and

vertical muscles). ~ The forward motion of the tongue is related to the pull of the posterior part of the genioglossus muscle, while the backward motion of the tongue is due to the contraction of the styloglossus. The posterior upward motion is a combination of stylo and palatoglossus muscles. The genioglossus muscle retracts and depresses the top of the tongue with the major fibers moving the tongue anteriorly. The hyoglossus muscle lowers the tongue. The vertical upward motion of the tongue is the result of the contraction of stylo and palatoglossus, whereas the downward motion is due to the action of the hyoglossus and the intermediate fibers of the genioglossus muscle. The concave formation of the tongue is a combination of styloglossus, palatoglossus and transversus function; the convex shape is related to the function of the hyoglossus. Central grooving of the tongue is a combination of transversus, vertical, styloglossus, and palatoglossus muscles. Spreading, flattening, and tapering of the tongue is predominantly a function of the verticalis muscle. The superior longitudinal muscle shortens the tongue, bulges, retracts and lifts the tip of the tongue, and constitutes the fastest articulator. The inferior longitudinal muscle shortens and depresses and retracts the tip of the tongue. This

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muscle is particularly important for the formation of the " S " sounds. The transversus muscle narrows, protrudes and grooves the tongue, the verticalis flattens and broadens the tongue. 3

Nerve Supply Sensation of the tongue is supplied by the trigeminal nerve through the lingual branch of the mandibular nerve. Taste fibers originate from the glossopharyngeal nerve and the chorda tympany fibers accompanying the lingual nerve. Both intrinsic and extrinsic motor functions derive from the hypoglossus nerve for the anterior two-thirds of the tongue. The palatoglossus muscle is innervated by the accessory nerve. The hyoglossus nerve runs deep in the anterior third of the tongue but becomes more superficial in the posterior portion and therefore is more vulnerable to operative damage in this position. Similarly, the lingual nerve is located in a deep position in the anterior portion of the tongue and becomes more superficial in the posterior part. Sensory function and taste are located primarily at the lateral part of the tongue, ranging from the tip to the back. Bitter taste is predominantly located at the back of the tongue within the vicinity of the papilla vallate. The taste of salt and sour is concentrated around the anterior and posterior edge of the mid-portion, whereas sweetness is predominantly located at the tip of the tongue. Taste receptors in the hard and soft palate, as well as in the pharyngeal mucosa, can compensate for the loss of sensory areas of the tongue. Most patients who have lost significant sensory-bearing parts of the tongue are therefore often not aware postoperatively of any lingual taste deficit. Deficit for sensation, supplied by the lingual nerve, is also rarely of clinical significance.

Speech Speech consists of an interaction of respiration, resonation, articulation, integration, and phonation. 2 The tongue is not only one of the most important articulators, but it also acts as a resonator, influencing the quality of vowels. Articulation consists of a combined function of supraglottic structures, modifying the breath stream and producing mainly unvoiced sounds. The resonation of vowels by the tongue, raised either in front or back, modifies the quality of

VELCEK ET AL.

the sounds. 2'4 Only a few vowels are produced with the tongue either low and flat or with the tongue raised both in front and back. Consonants do not follow this arrangement. The majority of consonants depends on the tongue's impedance of the air stream. The tongue is passive only in the formation of consonants like P, B, M, F, V, and H. 3 Consonants like L, N, T, and D are made with the tongue tip touching the same spot on the roof of the mouth. Other consonants are produced by the movement of the tongue against gums or teeth, to cause friction or plosion. In all speech problems, not only normal size and shape, but motility is essential. Since most speech difficulties involve S, Sh, and R sounds, mobility and coordination of the tongue movements can be evaluated by determining the quality of these articulated sounds. S is formed by the push of the tongue against the tooth ridge, whereas Sh is created when the back of the tongue is spread. Sigmatism (lisping), while also an indication of CNS damage, is a very sensitive indicator of motor control after surgery. One of the most subtle but valuable tests consists of checking the ability of the tongue to cleanse the inside of the upper molars, an indication of intraoral tongue mobility. More sophisticated methods of tongue evaluation consist of kymography, glossodynamometry, kalatography, xrays, and speech feedback. Mastication also depends on tongue mobility, with the tongue transferring food and mixing food particles and saliva. The tongue crushes food against the hard palate and then participates in the act of swallowing, simultaneously protecting the entrance into the larynx to prevent aspiration. Our experience with children in whom the epiglottis had to be resected showed that the normal functioning tongue alone is adequate to protect the larynx from aspiration during swallowing, after the epiglottis has been removed.

Relation of Operative Technique and Functional Deficit 5 13 The aim of operative intervention should be the preservation of speech, taste, sensation, size, shape, and mobility. As shown in Figs. 1, 2, and 3, there are several types of operative techniques: Vertical, central or marginal excisions, anterolateral pedicles and horizontal filleting. A

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241

VERTICAL

KOLE

CENTRAL

PICHLI:R EDGERTON

RHEINWALD

GUPTA

VERTICAL& CENTRAL

EGYI:DI OBCI:WI:SER

DI:PLAGNI:

LATERAL

DINGMAN GRAB

BUTLIN HANDLEY

location. Operative procedures leading to major damage of extrinsic muscles, such as the recommended transection of the genioglossus muscle in patients with a protrusion of the tip of the tongue 14 can interfere with important muscular function and should therefore be avoided. In patients with a deep or central lesion, the resection may lead to a major loss of the tongue; the preservation of lateral or antero-lateral pedicles (Fig. 2) can allow a functional reconstruction. Even though the tip of the tongue is then formed by the previous lateral part of the tongue, muscle function becomes surprisingly adaptable. Although the tip of the superior longitudinal muscle is now brought back to the side of the tongue, there is no recognizable major muscular deficit. It appears that the complicated interaction of intrinsic and extrinsic muscles can overcome the anatomic postoperative displacement of several muscle bundles. In patients with either macroglossia or diffuse neoplastic involvement, the reduction of the link of the tongue alone usually leads to a shortened but thick tongue with inadequate mobility. Hori-

MAGEE

Fig. 1. Vertical, vertical and central, and lateral glossectomias.

comparison of the anatomy of the tongue and the various operative procedures suggests that operative procedures limited to the anterior and superficial two-thirds of the tongue are the least likely ones to interfere with tongue function, sparing the lingual and hypoglossal nerve. These include the vertical and anterior wedge resections shown in Fig. 1. Central excisions are unlikely to endanger any neurogenic structures. If excessive, however, motor function of the verticalis muscle may be jeopardized. In patients with combined vertical and lateral excisions, as shown in Fig. 2, neurogenic injury can be avoided if the excision is limited to the upper two-thirds of the tongue in view of the deep position of the lingual and hypoglossal nerves. Marginal excisions are least likely to interfere with either nerves or essential muscles, but are also the least effective procedures if a uniform reduction of the tongue is planned. Excisions of posterior lesions, on the other hand, may expose either lingual, hypoglossal or glossopharyngeal nerves to injury in view of their more superficial

COMBINED

"~"

BECKER

ANTERO-LATERAL PEDICLE

(O.M.C.) Fig. 2.

Combined and antaro-lataral glossectomias.

242

VELCEK ET AL.

STAGED (D.M.C.)

\x.~ .-J

SHORTENING

THINNING Fig. 3. Staged vertical and horizontal glossectomies for primary and neoplastic macroglossia.

zontal filleting (Fig. 3) allows an extensive reduction of the tongue with the formation of a thin tongue with retained muscular mobility. Filleting of the tongue also avoids interference with the sublingual mucosa. A review of postoperative function I showed that reported mobility inteference ranged from 0% to 78%, the loss of mobility not only depending on the type of surgery and lesion, but also on the type of postoperative examination. The same author found, in his own group, a decrease of postoperative motility of 37% when the sublingual mucosa was involved in the operative procedure; if the sublingual mucosa was left intact, a motility decrease was limited to 20%. Sweet taste deficiency was present in 46% and salt taste deficiency in 19.2%. The taste for bitterness is unlikely to be compromised regardless of the operative procedure used. Although large central, as well as horizontal or lateral, excisions may interfere with taste, the patients are rarely aware of any taste loss postoperatively.

Congenital Tongue Lesions With the exception of macroglossia and lingual thyroid, major congenital defects are rare. ~5 They usually consist of a lower midline

cleft that may involve the mandible, the lower lip, and the tongue. This defect represents an anomaly of the lower half of the first branchial arch and only 26 cases have been reported in the literature. Non-neoplastic, primary macroglossia, although common, represents a problem that has led to many divergent opinions and approaches. Macroglossia can be primary or secondary. The secondary type, seen in children, consists of hemangiomas, lymphangiomas, neurofibromatosis, juvenile fibromatosis, or other infiltrating lesions. Primary congenital macroglossia not infrequently occurs in association with other syndromes, such as hyperthyroidism, glycogen storage disease, mongoloidism, cretinism, or amyloidosis.8 Unilateral macroglossia often occurs with other anomalies such as polydactyly, syndactyly or hemihypertrophy. ~~True congenital macroglossia is assumed to represent a hyperplasia with a numerical increase of muscle fibers, rather than hypertrophy. The definition of "macroglossia" is often difficult, since it may be relative, functional or structural. Some authors state that the failure of the tongue to fit behind the teeth establishes both diagnosis of macroglossia and indication for operative reduction. 7 Others felt that the atrophy of the papillae exposed to the air and occasional scars due to tooth biting can be an indication of the extent of the tongue to be resected. While some authors feel that a glossoplasty may be indicated in children with Beckwith syndrome, it is our feeling that operative intervention in true congenital macroglossia is only indicated if there is a major functional impairment. The frequency of articulatory errors decreases just before or during the first school year and then continues to decrease for 1 or 2 more years less rapidly. ~2 If speech impairment is considered to be the main indication for operation, the surgical intervention should therefore be delayed until the end of this period where spontaneous improvement ceases. Other authors s suggest an early repair of severe macroglossia before malocclusion occurs. Therefore, the indications for glossoplasty vary considerably. In our own experience, only 1 child out of 18 underwent a glossoplasty because of true congenital macroglossia. Yet in other reports u6, all giossoplasties were performed in patients with macroglossia only for the prevention or correc-

TONGUE LESIONS

tion of malocclusion or other orthodontic problems.

Lingual Thyroid Lingual thyroid, usually found in girls, can be complete or partially ectopic, or accessory. ~7'2~ The posterior part of the tongue, around the foramen cecum, is the most common location of the undescended thyroid. In a review of over 400 cases, ~8 90% were found within the tongue, the remaining 10% were found in the anterior neck superior to the hyoid bone. The diagnosis and location are confirmed by scintiphotography. Symptoms usually consist of dysphagia and dyspnea which also occurred in Patient 2, first seen at the age of 10 yrJ 9 The usual workup showed a single ectopic lingual thyroid in this patient. A review of the family history revealed that two other siblings had undescended thyroids located below the tongue. One of these children required thyroid supplement. Our patient, like the majority of patients reported 2~ responded well to thyroid medication. Only after the thyroid medication was accidentally cut in half at the age of 16 yr, did symptoms recur leading to an excision and pedicle implants. Postoperative scintigraphs failed to show function and the child is now on substitute thyroid therapy. The operative repair of a lingual thyroid, if necessary, can consist of either complete removal followed by thyroid hormone intake, or free transplantation or pedicle transfer. Pedicle transfer ~7-I9retaining a vascular pedicle, moving part of the thyroid into the neck, has been reported as successful on many occasions. Steinwald suggested that thyroid supplementation should not be given during or after surgery since it may decrease the chance of an operative S u c c e s s . 19 An angiogram prior to the operative procedure can identify the arterial blood supply of the lingual thyroid and be helpful in the development of a vascular pedicle if a neck transplant is attempted, j7 The free transplantation of the thyroid, usually done in 2-ram thick wafers, has been reported as successful in the abdominal wall, thigh, liver, and pectoralis muscle. ~8,2' While carcinoma of the lingual thyroid has been reported in 4% to 6% of all patients with lingual thyroids, it has not been reported in children under the age of 15 yr.

243

Cystic Lesions Solitary cystic lesions consisted of either salivary-type lesions or dermoids (teratoma). Minor salivary cysts, such as ranulas, were not included in this group. In patients with solitary cysts, radiographic contrast material was injected to outline the anatomic extension of the cyst. 22 While marsupialization is satisfactory in the treatment of a simple ranula, most intralingual cysts require excision. In one of our patients, Patient 3, a partial excision and marsupialization resulted in a satisfactory result. Of all dermoid cysts, 6.9% occur in the head and neck region, often in the floor of the mouth or the tongue and were first described by Butlin in 1885. 22 The differentiation between dermoid, epidermoid cysts, or teratoma follows that used in other anatomical locations. The location of the dermoid cyst is usually lingual or sublingual, often extending through the genioglossus or geniohyoid muscle. The congenital dermoid of the anterior one-half of the tongue is rare, however, and only a few have been reported. 22 In an occasional instance, the separation of the mylohyoid can lead to a dumbbell-shaped extraoral swelling as shown in one of our cases, Patient 4. The operative approach depends on the location, and can be either intraoral or transhyoid. A rare lesion, to be considered in a differential diagnosis, is the hydatid cystfl3 The cystic hygroma, or lymphangioma, represents the most common diffuse cystic lesion, usually occurring in infancy. It was first described by Virchow in 1 8 8 5 . 24 While the incidence of oral lymphangioma is not known, of 120 cases of cystic hygromas reported, 55 involved the neck, but only 3 occurred in the tongue and the sublingual space] 4 Inflammation of the tumor in this series occurred in 20%, in 7% repeatedly. Only two of these patients had associated hemangiomas but hemorrhage occurred in 12%. In our experience, cystic hygromas of the tongue are almost always associated with diffuse involvement of the floor of the mouth and the neck, and in two children it extended also into the pharynx and the perilaryngeal structures. The symptoms in all of our patients consisted of acute respiratory distress at birth, requiring immediate intubation and/or tracheostomy. All had marked salivation, indicating difficulty in

244

swallowing. The operative approach in our children consisted of staged resections for tongue, sublingual space, and neck. During the intraoperative interval, acute inflammation and accumulation of fluid and/or hemorrhage was common. Although hemorrhage into the cystic areas was common, the blood loss during the operative procedures was minimal and easily controlled. Complete removal of the lesions, however, was impossible in all cases. Repeat infection, sometimes severe, occurred up to 1 1 yr after the partial resection of the lesion. A 12yr-old follow-up in Patient 5 showed that the tongue has complete sensation and taste, good mobility with only minimal speech impairment in spite of some remaining tumor in the floor of the mouth (sigmatism and slight distortion of Sh sound if the child does not concentrate). In Patient 6, the tongue shape and posterior mobility is excellent, but the loss of the anterior lingual mucosa has led to a relative fixation of the anterior portion of the tongue requiring further mobilization. In contrast to others, we have not seen spontaneous regression of lymphangiomasfl4 In some patients, the differential diagnosis between pure cystic hygroma, hymolymphangioma and hemangioma can be difficult and may have to be proven by biopsy. While the operative repair of a pure hemangioma may occasionally be necessary, it was not necessary in our experience. If a resection is indicated, cryotherapy, electrocoagulation, or laser therapy may be helpful.27 In patients with predominantly capillary hemangiomatous involvement, the use of steroid therapy especially in early infancy is promising. Only in one of our patients with a diffuse oral facial hemangioma was repeated minor surgery indicated to repair recurrent sublingual granuloma pyogenicum, developing after repeat injuries to the sublingual hemangioma. Since the hemangioma was predominantly extralingual (sublingual, alveolar, and facial), we did not include this child in this series. Solid Tumors

Neurofibromas of the tongue can be localized or diffuse. The localized neurofibroma is often unilateral, s'9`~3,2s If macroglossia occurs in patients with neurofibroma, it is usually uni-

VELCEK ET AL.

lateral and the lingual nerve is often involved. 9 A loss of taste in a patient with a unilateral macroglossia may be the diagnostic clue of a lingual neurofibroma. Although they have been reported in children, they usually occur in the third or fourth decade of life29 and are usually seen in females. The site of this neurogenic tumor, as in Patient 10, is usually the posterior third of the tonguefl9 In a series of 28 lingual neurofibromas, 19 were in the back of the tongue, 4 were marginal and 3 were ventral. 29 Complete excision of solitary neurofibromas is not usually followed by recurrence. Occasional malignant degeneration of a neurofibroma has been reported. 28 Rapaport described a 4-yr-old boy with neurofibrosarcoma of the tongue, which was initially radiated, followed by surgery. Juvenile fibromatosis, known to occur in many parts of the body in children, in particular the supraclavicular area, has not been reported in the tongue according to our information. One of our patients (Patient 1 1) was born with a solid tumor at the junction of the anterior and middle third of the tongue extending across the midline. This lesion continued to increase disproportionately and proved to be juvenile fibromatosis at biopsy. At the age of 4 mo a partial but extensive glossectomy was performed and a left anterolateral pedicle was used to restore the tongue (Fig. 2). Although the left superior longitudinal muscle is now encircling almost the entire tongue, the muscular function has been excellent and the tongue mobility does not appear to be impaired or disturbed. An additional child with juvenile fibromatosis, involving primarily the pharynx, underwent two operative procedures in an attempt to eradicate the lesion, which proved futile. The local recurrence of the fibromatosis included the base of the tongue and finally led to the demise of the child. Since the primary lesion was extralingual, we have not included this child in our review. While there is no experience with radiotherapy of juvenile fibromatosis in the oral cavity, successful radiotherapy of lingual fibrosarcoma has been reported. 3~ Adult types of rhabdomyomas of the tongue have not been previously reported until Patient 9 was seen on our service and successfully treated by local resection. 3~Rhabdomyosarcomas, on the other hand, have been seen. 32 Thirty percent of

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245

all rhabdomyosarcomas in children are in the head and neck area, but only a few occur in the tongue. O f 68 children with rhabdomyosarcomas seen at the M a y o Clinic, 3 originated in the tongue. In 1 of these children, a 3-mo-old infant, excisional biopsy was followed with a recurrence. After the implantation of irridium seeds and regional chemotherapeutic infusion, the child remained asymptomatic without evidence of recurrence after a 5-yr follow-up. The child did, however, develop a hypoplasia of the mandible without primary detention. Granular cell myoblastomas, occurring in Patient 12, consist of predominantly benign neoplasms that usually occur within muscular tissue, although they can also arise in areas devoid of myoblasts. Granular cell myoblastomas are not infrequently found in the tongue in adults, but apparently rare in childhood. An 18-yr-old girl was reported 32 and the youngest child, according to our knowledge, is the 4day-old infant treated in our group. Although the lesion was large, it could be completely excised with a simple central glossoplasty. Other rarely found lesions that appear solid on physical

examination, include choristoma. 33,34

the oncocytoma 33 and

While carcinoma of the tongue is common in adults, it is rare in children and only a few have been reported. 35 37 The overall incidence of carcinoma of the tongue in young people under 21 yr of age is less than 3%. 36 Most carcinomas are squamous cell carcinomas, and adenocarcinoma, originating in minor salivary glands, has also been reported in childhood. 38 Minor tongue trauma, usually involving the tip of the anterior third of the tongue, is extremely c o m m o n in children but most repairs can be accomplished with local anesthesia alone. Only four children required the repair of extensive traumatic injury to the tongue, often associated with maxillofacial injuries. The repair, even in extensive tongue injury, is usually simple in view of the excellent vascularity of the tongue. Initial airway control, however, especially in children with combined facial maxillary injury, has not infrequently presented a major problem both in the emergency room and the operating room.

REFERENCES

1. Hardcastle WJ: Physiologyof Speech Production. New York, Academic Press, 1976, p 90 2. Diem CF: Introduction to the Anatomy and Physiology of the Speech Mechanisms. Springfield, Thomas, 1968, pp 143 146 3. Kaplan HM: Anatomy and Physiology of Speech (ed 2). New York, McGraw Hill, 1971, pp 361 362 4. Shearer WM: Illustrated Speech Anatomy (ed 2). Springfield, Thomas, 1968, pp 55-56 5. Bjuggren G, Jensen R, Strombeck JO: Macroglossia and its surgical treatment. Scand J Hast Reconstr Surg 2:116-124, 1968 6. Kriedler J, Rehrmann A: Tongue function after surgical reduction. Fortschr Kiefer Gesichtschir 18:266-272, 1974 7. Deplangne H, Mauchamp O: Effect of partial glossectomy on occlusionand facial growth. Compte Rendus Societe Francaise D'Orthopedie Dento-Faciale 46:133 138, 1975 8. Shafer AD: Primary macroglossia. Clin Pediatr 7:357, 1968 9. Hess J, Roed-Petersen K: A method of repair for unilateral macroglossia. Hast Reconstr Surg 59:439-442, 1977 10. Machtens VE, Schmallenbach HJ, Dieckhoff W: Semilateral macroglossia and its surgical treatment. Dtsch Zahnarztl Z 25:23-28, 1970 11. Merle-Beral Lorfeuvre: Symptomatology of macro-

glossias. Surgical indications for glossectomy. Rev Fr D'Odonto-Stomatologia 14:1641-1652, 1967 12. Arons MS, Solitare GB, Grunt JA: The macroglossia of Beckwith's syndrome. Hast Reconstr Surg 45:341 345, 1970 13. Gupta OP: Congenital macroglossia. Arch Otolaryngol 93:378-383, 1971 14. MacMillan HW: Unilateral vs. bilateral balanced occlusion. J Am Dent Assoc 17:1207-1221, 1936 15. Monroe C: Midline cleft of the lower lip, mandible, and tongue with flexion contracture of the neck. Case report and review of literature. Plast Reconstr Surg 38:312-319, 1966 16. Esser E, Loft P, Machtens E: Surgical correction of jaw abnormalities with special reference to the tongue. Fortschr Kiefer Gesichtschir 18:272-274, 1974 17. Huber P: Therapeutic plan in lingual goiter. Bruns Beitrage Zur Klinischen Chirurgie 214:71-80, 1967 18. Wertz ML: Management of undescended lingual and subhyoid thyroid glands. The Laryngoscope 84:507-521, 1974 19. Steinwald OP, Muehrcke RC, Economou, SG: Surgical correction of complete lingual ectopia of the thyroid gland. Surg Clin North Am 50:1177-1186, 1970 20. Katz AD, Zager WJ: The lingual thyroid. Arch Surg 102:582-585, 1971 21. Danis RK: An alternative in management of lingual

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thyroid: Excision with implantation. J Pediatr Surg 8:869870, 1973 22. Gold BD, Sheinkopf DE, Levy B: Dermoid, epidermoid and teratomatous cysts of the tongue and the floor of the mouth. J Oral Surg 32:107-111, 1974 23. Zaimi A: Hydatid cyst of the tongue. La Tunisie Medicale 53:303, 1975 24. Ninh TN, Ninh TX: Cystic hygroma in children. A report of 126 cases. J Pediatr Surg 9:191-195, 1974 25. Bozek J, Kozak-Bialasik D: Late results of surgical treatment of benign lingual tumors in children. Petiatria Polska 45:1193-1199, 1970 26. Jarzab G: Clinical experience in the cryosurgery of haemangioma. J Maxillofac Surg 3:146-149, 1975 27. Giunta J, Shklar G, McCarthy PL: Diffuse angiomatosis of the tongue. Arch Otolaryng 93:83 89, 1971 28. Rapaport A, deLima CP, Sobrinho J, et al: Neurogenic (neurofibrosarcoma) of the tongue. Int Surg 58:738739, 1973 29. Schenck P: A case report and discussion of the rare location of neurogenic tumors in upper respiratory and digestive regions. HNO 18:309-312, 1970 30. Nagy LT: Fibrosarcoma of the tongue. Int J Oral Surg 2:303-306, 1973

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