Macroglossia

Macroglossia A METHOD OF PLASTIC REPAIR RICHARD B. MAGEE,

From tbe Department Altoona, Pennsylvania.

of Surgery,

Altoona

Hospital,

ACROGLOSSJA is a reIativeIy rare condition. The etiologic factors are: (I) congenita1 muscular hypertrophy [1,2], (2) Iymphangioma and hemangioma [5], (3) myxedema and acromegaIy [?I, (4) glyconeogenesis (von Gierke’s disease), (5) tertiary syphilis [a], (6) primary and secondary amyloidosis [5,6], and (7) von Recklinghausen’s neurofibromatosis [7-y]. The dental impIications have been discussed by Boden and Pierce ]Jo] and Edgerton [II]. After appropriate medical measures, the surgeon is caIIed upon to reduce the size of the tongue without disturbing the basic anatomic structures. CaudeII and Yoshida [3] attempted a one stage operation, but suggested that two

M

FIG. I. Corona1 and nerves. A?7U:rican

Journal O/Surgery,

section

of tongue

Volume 103. May 1~62

Altoona, Pennsylvania

M.D.,

showing

stages might be Iess traumatic to the bIood suppIy of the tongue. To correct macrogIossia due to Iymphangioma in a five year old gir1, we devised and performed a two stage operation which seemed to be simpIe and effective. AnatomicaIIy, the dorsum of the tongue contains taste buds, mucous gIands and nerve endings. The main blood and nerve supplies are near the midline, in the lower haIf of the tongue muscuIature as viewed in the corona.1 section. (Fig. I.) In the first stage of the operation the length and width of the tongue are reduced. Measuring from the median sulcus with calipers, Keith needIes were thrust through the tongue 2.5 cm. from the midline. A continuous wedge of tissue was removed and hemostasis achieved.

Iines of excision

632

in reIation

to main vessels

Frc;. 2. Technic used in placing needles at proposed Iinc of excisiol~ in the vertical plane. The same method was in the horizotltal pl:rtle-excision in Stage 11.: uwti

FIG. 3. Sagittal section showing lines of wcisiondorsa1 to main arteries and nerves.

The needles \vere threaded with No. 2-o black silk, pulled through and tied. (Fig. 2.) In the second stage which is performed three months later, the thickness of the tongue is reduced. \\‘ith the tongue stretched anteriorly,

a wedge measuring 1.5 by 5 by 7 cm. \vas removed from anterior to posterior in the upper half of the tongue. A dorsal fIap measuring 1.5 cm. in thickness was preserved, as hvere the deep vessels and nerves. (Fig. 3.) Closure v’as

FIG. 4. Before operation.

FIG. 5. After operation. 633

Magee effected with interrupted No. 2-o bIack silk sutures. Postoperative care incIuded a cIear Iiquid diet for one week. The suture Iines were cIeansed with hydrogen peroxide and water every three hours to remove crusts and detritus. Photographs taken before and after operation are shown in Figures 4 and 5.

CASE REPORT D. D., a five year

old white

girl, was admitted

to

Altoona HospitaI on January 19, 1960. The chief compIaints were constant protrusion of the tongue and the inability of the patient to talk plainIy. The patient had been delivered at fuII term with no complications. Her development was as follows: sitting at three months, cutting lirst tooth at five months, saying “mommy,” “daddy” at six months and waIking at eight months. History indicated that she was aIlergic to spices and tomatoes, and had had no operations, accidents or serious illnesses. Family history showed no known mental disease. The patient had two younger siblings, both considered normal. On physica examination, temperature was 98°F.; puIse, I00 per minute; respirations, 20 per minute; blood pressure, 70/30-20 mm. Hg. Generally, the patient was small for her age, spoke infrequentIy and with garbIed speech. Skin was paIe with normal turgor. Facial features were “coarse,” with a flattened nose and epicanthal folds. Teeth were carious and the incisors were beginning to protrude. The tongue couId not be retained within the cIosed mouth. There were crusts, excoriations and Iarge papules measuring I to 2 mm. in diameter over the anterior and superior aspects. SaIiva dripped from the mouth continuously and the lips were dry and cracked. The thyroid gland was not paIpabIy enIarged. Deep reflexes were considered sIuggish but were equal bilateraIIy. Laboratory studies were as foIlows: hemoglobin, I I .6 gm. per cent; hematocrit, 37 per cent; white bIood ceI1 count, 6,561 per cu. mm.; buffy coat, 0.7. The differentia1 count showed 6 per cent eosinophils, 2 per cent banded neutrophiIs, 24 per cent segmented neutrophils, 66 per cent lymphocytes and 2 per cent monocytes. A catheterized urine specimen was yeIIow, cIear and neutral; specific gravity was 1.025; the urine was negative for aIbumin and sugar; there were o to 2 white bIood cells and 8 to IO red blood cells per high power. There were occasional uric acid crystaIs and mucus, with a small amount of amorphous urates. Urine cuIture was negative. TotaI choIestero1 was 189 mg. SeroIogic studies

for syphilis were nonreactive. Culture from the tongue showed: gamma streptococcus, coagulasepositive staphlococcus, and neisseria. No oral budding yeast celIs were noted. Protein bound iodine was 4.7 pg. Roentgenogram of the chest showed Iinear infiItration of the lower lobe of each lung, having the appearance of biIatera1 pneumonitis. Roentgenograms of the hands, wrists and elbows revealed that the bone age matched the chronologic age. Psychometric tests indicated that the patient’s menta1 age was equal to the chronoIogic age. On admission the diagnoses were macroglossia, gIossitis from exposure and infection, pneumonitis, cystitis and hypothyroidism which was later excIuded. Treatment for glossitis was frequent mechanica cleansing, antibiotics and nystatin (Mycostatin@). Cystitis and pneumonitis were controIIed. The administration of 25 pg. liothyronine (cytome1) twice daiIy was without benefit. On February 2, 1960, Stage I was performed and on February 12 sutures were removed, both with the patient under endotrachea1 anesthesia. The pathologic report was probabIe Iymphangioma (macroglossia lymphangiomatosum). Recovery was uneventfuh The patient was readmitted on May 26, 1960. At this time the patient was greatly improved. The mouth could be closed, gIossitis had disappeared, certain words were cIearIy understood and the patient was eating well. On May 28 Stage II was performed, with suture removal on June 7. Recovery was smooth and the patient was discharged on June g, 1960. At present there are no complaints and a process of retraining in speech is underway.

SUMMARY

I. The etiology of macrogIossia is reviewed and the anatomic considerations discussed. 2. A two stage operation for the correction of macrogIossia is presented and a case of Iymphomatous macrogIossia is reported. REFERENCES I.

2.

3. a.

5.

634

J. N. GongenitaI macrogIossia. Oh. &+Gynec., 7: 97, rgS6.BELL, H. G. and MILLAR, R. G. Congenital macrogIo&ia. Report of two cases. SW&~, 24: 125, ‘948. CAUDELL, W. S. and YOSHIDA, Y. K. Macroglossia in primary amyIoidosis. Surgery, 37: 643, 1955. ENSIGN. M. G. MacroaIossia as a manifestation of primary systemic agyloidosis. J. A. M. A., 149: 136, 1952. HENDRICK, J. W. MacrogIossia or giant tongue. Surgery, 39: 674, 1956. PANNULLO,

Macroglossia presenting as macrogIossia. Arch. Dis. Child., 26: 76, ‘95’. g. BIRD, 7‘. and CLARK, G. 0. Neurofibromatous macroglossia. J. Laryng. CYOtol., 69: 5 I, 1955. 10. BADEN, E. and PIERCE, il.E. Angular chrilosis and macroglossia. Oral Surg., 8: 100, 195 j. 1 I. EDGERTON, Xl. T. L’lanagement of’ macroglossia when associated with prognathism. Bsir. J. Plastic Surp., 3: 117, 1950.

6. R~c;~ou, R. t1. and NOBLIP;, F. E. 1Iacroglossia accompanying primary systemic amyloidosis. .4nn. Otol., Kbin. 0” Lqng., $8: 470, ‘04% 7. Auu, hl. hl., DELAMI, A. J. and BACKEK, h,I. H. Congenital ncurofibromatosis macroglossia associated in some cnsvs with van Recklinghausen’s disease. Cancer, 5: 721, I9$2. 8. Kr \,PTON, .I. J. and LATTO, C. Neurofibromatosis

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