Leukoedema of the buccal mucosa in Negro children and youth James L. Martin, DEPARTMENT
D.D.S.,”
OF PEDIATRICS,
and E. Perry MEHARRY
Crump, M.D.,“”
MEDICAL
Nashville,
Tenn.
COLLRGE
Leukoeclema has been reported to occur only in adult populations. In 1953, Sandstead and Lowe suggested that there may be a tendency for leukoplakia or other keratoses to develop in areas of leukoedema. It was further stated that there is a definite predilection for this lesion in the Negro. The etiology is unknown. In the present investigation the buccal mucosae of 1,000 Negro children and teen-aged patients were examined for leukoedema. An incidence of 50.8 per cent was observed. Histopathologic examination of biopsiecl leukoedematous specimens failed to demonstrate any features suggestive of dyskeratosis or carcinoma.
L
eukoedema, a grayish white lesion which characteristically appears on the oral buccal mucosa, has been alleged to occur only in adult populations1 The cause is unknown. Sandstead and Lowe2 examined 646 patients and found the condition in 43 per cent of the Caucasian and 90 per cent of the Negro patients. They concluded that this disease has a definite predilection for the Negro. “It was suggested that leukoedema may represent a lesion of the mucosa in which leukoplakia or other keratoses are more likely to develop than in normal epithelium.“2 Pindborg and colleague& 4 reported this lesion in 96 of 10,000 patients observed in Bombay, India, and in 166 of 10,000 Indians in Lucknow. Martin, Buenahora, and Bolden found leukoedema in 203 of 300 hospitalized male and female Negro patients. The purposes of the present investigation were to determine the incidence of leukoedema in Negro children and to test the leukoedemic occurrence for possible correlation with age, sex, oral hygiene status, syphilis, malocclusion, occurrence and degree of melanin pigmentation of the buccal mucosa, and occurrence in siblings. Presented at the forty-ninth annual meeting of the Research, Chicago, Ill., March, 1971. *Assistant Professor and Director of Dental Services. **Professor, Chairman, and Project Director.
International
Association
for
Dental
49
Oral July,
Fig.
1
Pig. 1. Slight leukoedema in a G-year-old Negro boy. of the buccal mucosa. Fig. 8. Moderate Icukoedenm in a 15year-old Negro delineates the lesion.
Fig. 3. Severe middle and posterior
MATERIALS
Surg. 1972
AND
leukoedema in a 15.year-old areas of the buccal mucosa.
Negro
boy.
Note girl.
Note
t.he faint Note
tho
the
grayish upper
radiating
white
d
area
border
which
striac
in the
METHODS
The buccal mucosae of 1,000 Negro children were examined with artificial light and a mouth mirror. The occurrence and degree of leukoedema were assessed according to the criteria previously defined by Sandstead and Lowe2: Slight-Mucosa was opalescent and thickened, with the capillary network obliterated; the entire mucosa may be involved but the middle and posterior portions of the cheek were principally affected; the edematous appearance disappeared upon stretching. Moderate-Mucosa had a coarsely granular surface with a definite white or whitish gray appearance that persisted upon stretching.
Volume 34 Number1
Leukoedema
of buccaZmucosa in Negroes
51
28.5496 44.6% 26.7%
I 50
1 0
1 0
I 2 0 250
3 0
3 0
I 400 4$0
5bo
5bo 40
80
NUMBER OF CASES
Fig. 4. Incidence of leukoeclema.
Severe-Mucosa appeared grayish white and thickened; in some cases it appeared shaggy and wet, and in other instances it was leathery and dry (Figs. 1, 2, and 3). The buccal mucosa was anesthetized by topical application of 5 per cent lidocaine ointment. Biopsy specimensof the leukoedematous tissues were fixed in 10 per cent formalin, sectioned, and stained with hematoxylin and eosin. The occurrence and degree of melanin pigmentation of the buccal mucosa were assessedas “none,” “slight,” “moderate,” or “heavy.” This determination was arbitrarily based on the presence or absenceof visible melanin pigmentation. RESULTS Population
studied
A random selection consisting of 1,000 Negro patients registered in the Meharry Comprehensive Health Care Program for Children and Youth was made for this study. The series consisted of 501 boys and 499 girls ranging in age from 1 to 18 years, with an average age of 9.2 years. Incidence
Five hundred eight (50.8 per cent) of the subjects had leukoedema of the buccal mucosa. The condition was generally observed bilaterally. “Slight” leukoedema was found in 145 (28.54 per cent) of the cases,whereas “moderate” and
52
Martin
Oral July,
and Grump
53 15
Surg. 1972
- ‘al,. -,,-
I
Fsmrl,?
I 2
II 3;
I 5
II 67
I d
I 3
10
III
11
12
I 13
II 1-l 15
16
I
II 17
I?
AGE IN YEAS
Fig. 5. Incidence
of leukoedema
in relation
to age and sex.
“severe” leukoedema were found in 227 (44.69 per cent) and 136 (26.77 per cent), respectively (Fig. 4).
The youngest patient with leukoedema was 3 and the oldest was 18 years of age. The majority of casesof leukoedema was observed in the 13- to 18-year age group, but the condition was most prevalent in U-year-old boys and in 12- and 15-year-old girls (Fig. 5). A direct relationship between the severity of leukoedema and age groups was demonstrated (Fig. 6). Sex Two hundred sixty-eight boys (52.76 per cent) and 240 girls (47.24 per cent) exhibited leukoedema. There was no sex predilection (Fig. 4). Oral
hygiene
status
Although a definite relationship between leukoedema and the variables of excellent, good, and fair oral hygiene was not evident, a direct correlation between the occurrence of leukoedema and poor oral hygiene was established (Fig. 7).
Volume Number
Leukoedema
34 1
lOO-
0
Slight
95-
69 m
Moderate Severe
of buccal mucosa in Negroes
53
9085807570 6560555045-
AGE GROUPS
Fig.
6. Severity
of leukoedema
in relation
to age groups.
Serology
Negative Wassermann reactions were reported for all the children comprising this study. Malocclusion
Four hundred twenty-three subjects were examined for possible correlation between leukoedema and malocclusion. Forty-two patients presented with both leukoedema and malocclusion, whereas 154 children exhibited leukoedema and normal occlusion. In comparison, 187 patients of this study had normal buccal mucous membranes and normal occlusion. Forty patients had malocclusion and normal buccal mucosae. Melanin
pigmentation
A direct relationship between the severity of leukoedema and the degree of melanin pigmentation of the buccal mucosae was noted. Two patients of this series did not have pigmented buccal mucous membranes and did not have leukoedema. Of the 155 patients with slight pigmentation of the buccal mucosae, 25
Oral July,
Surg. 1972
260240-
160
60
Excellent
Good ORRL HYGIENE STATUS
Fiy.
7. The
relation
of leukoedema
and oral
hygiene.
(16.13 per cent) exhibited leukoedema. Leukoedema was present in 376 subjects with moderately pigmented buccal mucosae (51.62 per cent) and in 116 (87.88 per cent) of those children with heavily pigmented buccal mucosae (Table I). Oral
habits
The buccal mucosae of 383 subjects were examined for possible correlation between the presence of leukoedema and the oral habits of finger-sucking, thumbsucking, tooth-grinding, cheek-biting, and tongue-sucking. Of the 182 children who exhibited leukoedema, 36 patients had an oral habit. One hundred forty-six of these subjects did not have abnormal oral habits. Siblings
The occurrence of leukoedema among siblings is not directly related. All the siblings of thirty-five families exhibited leukoedema, whereas all the children of forty families presented with either leukoedema or normal buccal mucosae. Thus, some of the children of the same family exhibited the condition while others did not (Table II). Histologic
findings
Selections of biopsied leukoedematous tissue from thirty patients revealed essentially the same characteristics as previously reported in an adult popula-
Leukoedema
Fig. 8. Note the group of irregular darker area in the upper right-hand (Hematoxylin and eosin stain. Magnification,
Table
of buccal mucosa in Negroes
leukoedematous portion of x16.)
this
cells near the surface. The photomicrograph is an
55
circular, artifact.
I. The relation of leukoedema and melanin pigmentation
Degree of melanin
Heavy Moderate Slight Negative
132 711 155 2
Tots1
Table
116 367 25 0 508
1,000
87.88 51.62 16.13 0
12.12 48.38 83.87 100
3:: 130 2
100 100 100 100
492
II. The occurrence of leukoedema in siblings ~ Number 1 1
Conddion of buccal mucosa Leukoedema Normal Leukoedema normal
of siblings 3
(
4
per family 1
5
1
6
Number families 35” 40t
26 37
6 2
2 0
1 2
0 0
19
13
6
5
1
of
Number children
of Per
ii
cent 27.67 28.33
or 44t
Total
132
119 *Number
of families
in which
all siblings
had leukoedema.
tNumber
of families
in which
all siblings
had normal
$Number
of
families
in which
some
siblings
exhibited
44.00
300
buccal
100
mucosae.
leukoedema
while
others
did
not.
tion by Martin and associates6: (1) Th ere was intracellular edema of the Malpighian layer. (2) Leukoedematous cells assumedirregular polyhedral shapes. (3) Vacuolization of the cytoplasm was quite common; the vacuoles appeared to contain a pale-staining homogeneous fluid. (4) There was a wide variation in nuclear size, and the contained chromatin material was either irregularly condensed, eccentrically positioned or completely absent (Figs. 8,9, and lo),
56
Martin
Pig. Malpighian x16.) Fig. vacuolated
and
9. Note layer
Grump
the eccentrically positioned extend to the basal-cell layer.
10. High-pan-er and enucleated
pyknotic nuclei. Leukoedematous (Hematoxylin and eosin stain.
viem of an area from the specimen shown in Fig. cells. (Hematoxylin and eosin stain. Magnification,
cells in Magnification,
0. Note x40.)
the
the large
DISCUSSION Leukoedema of the buccsl mucosa was previously thought to occur only in adult populations. An incidence of 50.8 per cent was observed in the children and teen-agers comprising this study. This condition, of unknown etiology, generally occurred bilaterally and appeared to have no sex predilection. The youngest child with this clinical entity was 3 years of age. ‘(Severe” leukoedema was found to occur only in children 5 years of age or older. The severity of leukoedema appeared to increase with age. The highest incidence was observed in U-year-old boys and 12- and 15-year-old girls. The high percentage of cases of “moderate” severity may represent a transitory phase between “slight” and “severe” stages. A previous investigation by
Leukoederna
of buccal mucosa in Negroes
57
Martin and associate@ revealed a low percentage of “moderate” cases of leukoedema in Negro adults. This apparent inverse relationship could be attributed to progressive changes of this condition from “slight” to “moderate” in the earlier years of the patient’s life, since the severity of leukoedema increased with age. The high incidence of leukoedema in children with poor oral hygiene suggests that poor oral hygiene may play a supportive role in its etiology. Syphilis, malocclusion, and oral habits were not causative factors. A direct relationship between the severity of leukoedema and the degree of melanin pigmentation of the buccal mucosa was observed. Leukoedema appears to be a condition that is manifested in the buccal mucosa of the patient; some siblings exhibit this entity while others have perfectly normal buccal mucosae. Leukoedematous tissues of the buccal mucosae of Negro children and teenaged patients gave the same clinical and histologic response that was previously reported in Negro adults.G The primary histologic characteristic of leukoedema and the key to its etiology may be centered in those cells which become edematous, vacuolated, and eventually enucleated. Although the etiology of leukoedema is unknown, future investigations should encompass children and teen-aged populations. SUMMARY
AND
CONCLUSIONS
A study was made of the incidence of leukoedema of the buccal mucosa in 1,000 Negro children and teen-agers. 1. Leukoedema of the buccal mucosa occurred in Negro children and youth. An incidence of 50.8 per cent was observed in this study. 2. Leukoedema occurred in a 3-year-old child, but the highest incidence was observed in U-year-old boys and 12- and 15-year-old girls. 3. The severity of leukoedema increased with age. 4. There was no sex predilection. 5. Syphilis, malocclusion, and oral habits did not appear to be causative factors. 6. A definite relationship between leukoedema and poor oral hygiene was established. 7. The severity of leukoedema was directly proportional to the degree of melanin pigmentation of the buccal mucosa. 8. The large number of “moderate” cases suggests a transitory stage between “slight” and “severe.” 9. No relationship between leukoedema and its occurrence in siblings was observed. 10. Histologically, leukoedema was defined as a benign entity with no features of dyskeratosis or malignancy. REFERENCES
1. Archard, H. O., Carlson, K. P., and Stanley, H. R.: Leukoedema of the Human Oral Mucosa, OFCALSUEW. 26: 717-728, 1968. 2. Sandstead, H. R., and Lowe, J. W.: Leukoedema and Keratosis in Relation to Leukoplakia of the Bucca.l Mucosa in Man, J. Natl. Cancer Inst. 14: 423-437, 1953.
Oral July, 3. Pindborg, J. J., Kalapessi, II. K., Kale, d. A., Singh, G., and Talyerkhan, Frequency of Oral Leukoplakias and Related Conditions Among 10,000 Bombayites, India Dent. Assoc. 37: 1102, 1965. 4. Pindborg, 5. J., Chawla, T. N., Misra, R. I~., Nagpaul, R. K., and Gupta, B. K.: and Lichen Planus in 10,000 lndians in Lucknow, Uttar Pradesh, India, J. Dent. 615, 1965. 5. Martin, 5. L., Buenahora, A. M., and Bolden, T. E.: Leukoedema of the Bnccal The Meharri-Dent. 29: 7-9, 1970. 6. Martin, J. L., Buenahora, A. M., and Bolden, T. E.: Cyto-histology of Leukoedema Buccal Mucosa, J. DC Dent. Sot. 44: 47-52, 1969. Reprint requests to: Dr. James L. Martin School of Dentistry Meharry Medical College 1005 Eighteenth Ave., N. Nashville, Tenn. 37208
Surg. 1973 B. N.: J. All
Fibrosis Res. 44: Mucosa, of the