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Lichen planus or leukoplakia?
Lichen planus or leukoplakia? A case
of clinical
interest
L
ichen planus is an inflammatory dermatosis affecting the skin and mucous membranes. In the mouth it may appear in several different patterns’, ‘); one of the most confusing is that in which greatly increased keratinization occurs, forming thickened elevated plaques. It, would seem to be of interest to present a case in which a striking clinical change occ*urred in the mouth of a patient with long-standing lichen planns and to consider the significance of this alteration. CASE
REPORT
In 1965 a Si-year-old engineer of Eastern Europ
011 c*liniCill
r9camin:ltion,
tht> dcntitiou
corwihxl
of
-.----I_and ttlc, missing 5132111234 teeth had ~WWI replaced with satisfactory partial dwtuws. Oral hygiene WRR generally good. Typicsal lesions of lichen planus were seen on 111~ tongue (atrophic pattrm), gingiw (reticulated pattern), and bilaterally on the cheeks (annular patterns) (Fig. 1). 1 n the 16t axilla a l~lur rash, composed of pxpules, was noted, and this was considered to be part of tlifb same dermatosis that affected the mouth. The hemoglobin level was found to be 103 per rent, and the white blood cell count, was 7,300 per cubic centimeter with a normal differential. The Wassermann and Kahn test ream tions were negative. No biopsy of any of the lesions was performed, as t,he clinical pwsenta~ tion was considered to be characteristic of lichen planus.
850
Eggleston
Oral Surg. June, 1970
Fiy.
1. Lesion
of lichen
Fig.
8. Plaquelike
lesion
planus
seen in right
seen in right
buccal
buccal
mucosa
mucosa
in 1965.
in 1969.
Treatment was commenced with triamcinolone 0.1 per cent in a mucous-membrane-adherent ointment, and this resulted in a rapid diminution of the symptoms together with some gradual regression of the lesions. The skin rash eventually cleared completely and has never recurred. In 1966 the patient developed a bullous lesion in the left cheek, which rapidly broke down to give the appearance of erosive lichen planus. Local steroid preparations quickly healed the ulceration, and the patient was not seen for two years. In 1968 the patient returned with ulcerations on both cheeks and on the gingivae. A further course of topical steroids in emollient dental base was required to produce healing. It was at a follow-up clinic in March, 1969, that a small white thickening was noted in the anterior region of the right cheek above the occlusal line. When seen two months later, this roughened area was considerably larger, with a keratotic white surface which could not be removed by rubbing or scraping; it presented the appearance of what is usually clinically
Volume Number
Lichen
29 6
F1:g. 3. Histology
of
original
planus
or leukoplakia?
851
lesion.
described as ‘Lleukoplakia” (Fig. 2). The original lesions of lichen planus mere noted to bts still present in the mouth. A review of the medical history of the preceding five years revealed no significant alteration in the patient’s way of life. He was still a uonsmoker who drank only occasionally and whose social circumstances appeared unchanged. His dentures had not been remade, and examination showed them to be reasonably satisfactory ; certainly, there were no sharp edges or clasps to act as a mucosal irritant. The hemoglobin level was 103 per cent, and the white blood cell count was 7,200 1~ cubic centimeter with normal differential. No lupus erythematosus cells were found, and iiuh serum protein levels were within the normal range. Tests for syphilis were negative, and ncl evidence of candidiasis could be demonstrated. A biopsy of the keratotic area was performed xud the report stated that the lesional epithelium was partially orthokeratinized and partially hyperkeratinized. There was acanthosis, and many prickle cells exhibited individual cell keratinization. A fairly diffuse inflammatory cell infiltration of mixed type was present in the underlying corium. The appearances were not those of a typical lichen planus; they were consistent with those of “leukoplakia.‘? (See Fig. 3.) In view of the rapid growth of the keratotic area, its relatively small size, and the ease of access, total excision of the lesion was performed under local analgesia. The opportunity was taken to perform a biopsy on a typical area of the original lichen planus. This was reported as characteristic of lichen planus (Fig. 4).
DISCUSSION
This case report describes a patient in whom two variations of hyperkeratcrsis occurred in the mouth. In view of t,heir apparent dissimilarity, it is reason-
052
Oral Surg. June, 19X
Eggleston
Fig. 4. Histology
of plaque lesion.
able to consider the possibility that the different clinical appearances may represent separate pathologic conditions. There is undoubtedly a form of lichen planus which bears a close clinical resemblance to “leukoplakia,” and it may be extremely difficult to separate the two conditions, particularly if histologic study of the lesion shows it to be nonspecific. However, a careful search at the periphery of the plaque may reveal individual papules of lichen planus which will help in making the diagnosis.” Nevertheless, several authors4-g have described cases in which the clinical and histologic features characteristic of lichen planus and “leukoplakia” were present together in the mouth of one patient. Unfortunately, for many years the term leukopl&Ga has been used to describe any mucosal lesion with the appearance of a white plaque and, as a result, the term has been used by different observers to describe varying disorders. CookelO preferred the term kemtosis to leukopl&ia and divided cases, according to their etiology, into smoking, frictional, syphilitic, and idiopathic are now recoe-nized RR sensmt@ olin;~~l nn+:+;no ---l--n--groups. These divisions
Volume 29 Number 6 previously they were all described under the title ZeukopZa7cia; thus, some of the cases reported with a double diagnosis of lichen planus and leukoplakia might more correctly be represented as disorders of smokers’ keratosi$ or frictional keratosis5j 8 and lichen planus. Other reports do not give sufficient dctails to allow such accurate descriptions to be used, and they may represchnt examples of idiopathic keratosis and lichen planus occurring together. In this article, leukoplakia is considered to be any white patch or plaque on the oral mucous membrane that cannot be removed by scraping, cannot be reversed by the removal of obvious irritants, and cannot he classified clinically or microscopically as another diagnosable disease. ‘I It, is, thcrefonc, a diagnosis made by exclusion and, in Cooke’s classification, would form part of that iciiopathic group. The difficulty with those cases of leukoplakia as defined in this articltl is that they are sufficiently common in occurrence to present problems in mar~;rgt~mcnt, for the condition is known to be precancerous in nature. According to Silverman,ll this latter statement is based on the finding of a significant. ~tnmher of oral carcinomas with associated regions of leukoplakia and the fact, that oral leukoplakia does precede some eventual malignant. epithelial t ransfornmtion. Silverman also quotes several series of investigations into Icukoplakia in which the rate of malignant change varies from 11 to 60 per cent. It is not so well known that lichen planus may also become malignant, but an increasing number of cases are being reported in the literature. Andrea~son” reported references to forty-six such previously recorded cases, and in eleven of them carcinoma had developed in plaquelike lesions. The rate of malignant change has been reported to be as high as 10 per cent in separate series by Dechaumelz and Warin. More recently,l” it has been stated t,hat t,he over-all malignancy rate in oral lichen planus is probably less than I per cent, but even at this rate the incidence is possibly greater than t,hc random chance assocG1tion of carcinoma with a relatively common disorder. In a discussion of lichen planus as long ago as 1931,1* emphasis was placed on the importance of differentiating the gross appearance of this condition from moniliasis. With the introduction of antibiotics, steroids, and cytotoxic drugs7 candidiasis of the mouth is a much more frequent occurrence, and it is now recognized that there exists a chronic hyperplastic formI in which thcl rvhite plaque cannot be removed by scraping. The histopathology in t,hcse cases is specific, but clinically the thick, edematous epithelial plaques are indistinguishable from “leukoplakia.” Recently, there has been a report of thirty wws oi candidial “leukoplakia” which were followetl for ‘L pears; in that time, nine underwent, malignant change.13 The patient whose case has been presented here was a nonsmoking, 1101~. syphilitic man with a dentally fit mouth; in the absence of mechanical, microbial, or fungal influences and, in view of the varying histology, it is impossible to account for the sudden appearance of the keratotic area observed in the mout-h or to state whether this represents only a variation of the lichen planus already present. Nevertheless, after consideration of the sinister clinical appearance and rapid growth of the lesion, and in view of the unpredictable occurrcncc 01’
854
Eggleston
Oral Surg. June, 1970
malignant conditions arising from apparently benign lesions of leukoplakia,ll total excision was adopted as the most satisfactory treatment in this case. The interest of this communication is that, although a consideration of the etiology has provided the grounds for a discussion point, it is the diagnosis at the clinical level which has formed the basis of treatment. Rather than subject the patient to repeated hospital admissions for periodic observation of the lesion, it was decided to remove the keratotic area completely and thus reduce to a minimum any risk to which the patient might have been exposed. SUMMARY A case of long-standing lichen planus in which a plaquelike lesion developed in the oral mucosa is reported. The significance of this lesion is discussed with regard to possible malignant change. Surgical removal is considered the most suitable form of treatment for all plaquelike lesions of the oral mucosa if they are of relatively small size. I wish to thank Professor Sir Robert Bradlaw for permission to use his cases and Mr. F. Nally for his encouragement and help with this paper. I would also like to thank the personnel of the Pathology Department and the Photographic Department of the Eastman Dental Hospital for their kind assistance and advice. REFERENCES 1. Andreason,
J.: Oral Lichen Planus, ORAL SURG. 25: 31, 1968. of Lichen Planus, Brit. Dent. J. 96: 1, 1954. 2. Cooke, B. E. D.: Oral Manifestations 3. Darling, A. I., and Crabb, H. 5. M.: Lichen Planus, ORAL Suaa. 7: 1276, 1954. Brocq, Q. J.: Lichen Plan des muqueuses et leuco plasies, Presse med. 27: 277-279, 1919. D., and Culver, G.: Lichen Planus of the Mouth Alone, Brit. J. Derm. 41: i: Montgomery, 45, 1929. 6. Wolf, J.: Lichen Planus and Leukoplakia, Arch. Derm. (Chicago) 63: 805, 1951. 7. Altman, J., and Perry, H.: Variations and Course of Lichen Planus, Arch. Derm. (Chicago) 84: 179-191, 1961. 8. Warin, R.: Epithelioma Following Lichen Planus in the Mouth, Brit. J. Derm. 72: 288, 1960. 9. Andreason, J., and Pindborg, J.: Canceruduiklug i oral lichen planus, Nord. Med. J. 70: 861-866, 1963. 10. Cooke, B. E. D.: Leukoplakia Buccalis and Oral Epithelial Naevi, Brit. J. Derm. 68: Xl174, 1956. 11. Silverman, S., Renstrup, G., and Pindborg, J.: Studies in Oral Leukoplakia, Acta Odont. Stand. 21: 271, 1963. 12. Dechaume et al.: Le lichen plan isole de la muquesse buceali, Presse med. 65: 2133-2135, 1957. 13. Association of Head and Neck Oncologists of Great Britain, Premalignant Lesions Affecting the Mouth, Pharynx and Larynx, Brit. Med. J. 2: 570, 1969. 14. Fox, H.: Lichen Planus Confined to the Mouth, Arch. Derm. (Chicago) 24: 1071-1082, 1931. 15. Lehner, T.: Oral Thrush in Acute Pseudo Membranous Candidiasis, ORAL SURG. 18: 27,
1964.
of clinical
interest
L
ichen planus is an inflammatory dermatosis affecting the skin and mucous membranes. In the mouth it may appear in several different patterns’, ‘); one of the most confusing is that in which greatly increased keratinization occurs, forming thickened elevated plaques. It, would seem to be of interest to present a case in which a striking clinical change occ*urred in the mouth of a patient with long-standing lichen planns and to consider the significance of this alteration. CASE
REPORT
In 1965 a Si-year-old engineer of Eastern Europ
011 c*liniCill
r9camin:ltion,
tht> dcntitiou
corwihxl
of
-.----I_and ttlc, missing 5132111234 teeth had ~WWI replaced with satisfactory partial dwtuws. Oral hygiene WRR generally good. Typicsal lesions of lichen planus were seen on 111~ tongue (atrophic pattrm), gingiw (reticulated pattern), and bilaterally on the cheeks (annular patterns) (Fig. 1). 1 n the 16t axilla a l~lur rash, composed of pxpules, was noted, and this was considered to be part of tlifb same dermatosis that affected the mouth. The hemoglobin level was found to be 103 per rent, and the white blood cell count, was 7,300 per cubic centimeter with a normal differential. The Wassermann and Kahn test ream tions were negative. No biopsy of any of the lesions was performed, as t,he clinical pwsenta~ tion was considered to be characteristic of lichen planus.
850
Eggleston
Oral Surg. June, 1970
Fiy.
1. Lesion
of lichen
Fig.
8. Plaquelike
lesion
planus
seen in right
seen in right
buccal
buccal
mucosa
mucosa
in 1965.
in 1969.
Treatment was commenced with triamcinolone 0.1 per cent in a mucous-membrane-adherent ointment, and this resulted in a rapid diminution of the symptoms together with some gradual regression of the lesions. The skin rash eventually cleared completely and has never recurred. In 1966 the patient developed a bullous lesion in the left cheek, which rapidly broke down to give the appearance of erosive lichen planus. Local steroid preparations quickly healed the ulceration, and the patient was not seen for two years. In 1968 the patient returned with ulcerations on both cheeks and on the gingivae. A further course of topical steroids in emollient dental base was required to produce healing. It was at a follow-up clinic in March, 1969, that a small white thickening was noted in the anterior region of the right cheek above the occlusal line. When seen two months later, this roughened area was considerably larger, with a keratotic white surface which could not be removed by rubbing or scraping; it presented the appearance of what is usually clinically
Volume Number
Lichen
29 6
F1:g. 3. Histology
of
original
planus
or leukoplakia?
851
lesion.
described as ‘Lleukoplakia” (Fig. 2). The original lesions of lichen planus mere noted to bts still present in the mouth. A review of the medical history of the preceding five years revealed no significant alteration in the patient’s way of life. He was still a uonsmoker who drank only occasionally and whose social circumstances appeared unchanged. His dentures had not been remade, and examination showed them to be reasonably satisfactory ; certainly, there were no sharp edges or clasps to act as a mucosal irritant. The hemoglobin level was 103 per cent, and the white blood cell count was 7,200 1~ cubic centimeter with normal differential. No lupus erythematosus cells were found, and iiuh serum protein levels were within the normal range. Tests for syphilis were negative, and ncl evidence of candidiasis could be demonstrated. A biopsy of the keratotic area was performed xud the report stated that the lesional epithelium was partially orthokeratinized and partially hyperkeratinized. There was acanthosis, and many prickle cells exhibited individual cell keratinization. A fairly diffuse inflammatory cell infiltration of mixed type was present in the underlying corium. The appearances were not those of a typical lichen planus; they were consistent with those of “leukoplakia.‘? (See Fig. 3.) In view of the rapid growth of the keratotic area, its relatively small size, and the ease of access, total excision of the lesion was performed under local analgesia. The opportunity was taken to perform a biopsy on a typical area of the original lichen planus. This was reported as characteristic of lichen planus (Fig. 4).
DISCUSSION
This case report describes a patient in whom two variations of hyperkeratcrsis occurred in the mouth. In view of t,heir apparent dissimilarity, it is reason-
052
Oral Surg. June, 19X
Eggleston
Fig. 4. Histology
of plaque lesion.
able to consider the possibility that the different clinical appearances may represent separate pathologic conditions. There is undoubtedly a form of lichen planus which bears a close clinical resemblance to “leukoplakia,” and it may be extremely difficult to separate the two conditions, particularly if histologic study of the lesion shows it to be nonspecific. However, a careful search at the periphery of the plaque may reveal individual papules of lichen planus which will help in making the diagnosis.” Nevertheless, several authors4-g have described cases in which the clinical and histologic features characteristic of lichen planus and “leukoplakia” were present together in the mouth of one patient. Unfortunately, for many years the term leukopl&Ga has been used to describe any mucosal lesion with the appearance of a white plaque and, as a result, the term has been used by different observers to describe varying disorders. CookelO preferred the term kemtosis to leukopl&ia and divided cases, according to their etiology, into smoking, frictional, syphilitic, and idiopathic are now recoe-nized RR sensmt@ olin;~~l nn+:+;no ---l--n--groups. These divisions
Volume 29 Number 6 previously they were all described under the title ZeukopZa7cia; thus, some of the cases reported with a double diagnosis of lichen planus and leukoplakia might more correctly be represented as disorders of smokers’ keratosi$ or frictional keratosis5j 8 and lichen planus. Other reports do not give sufficient dctails to allow such accurate descriptions to be used, and they may represchnt examples of idiopathic keratosis and lichen planus occurring together. In this article, leukoplakia is considered to be any white patch or plaque on the oral mucous membrane that cannot be removed by scraping, cannot be reversed by the removal of obvious irritants, and cannot he classified clinically or microscopically as another diagnosable disease. ‘I It, is, thcrefonc, a diagnosis made by exclusion and, in Cooke’s classification, would form part of that iciiopathic group. The difficulty with those cases of leukoplakia as defined in this articltl is that they are sufficiently common in occurrence to present problems in mar~;rgt~mcnt, for the condition is known to be precancerous in nature. According to Silverman,ll this latter statement is based on the finding of a significant. ~tnmher of oral carcinomas with associated regions of leukoplakia and the fact, that oral leukoplakia does precede some eventual malignant. epithelial t ransfornmtion. Silverman also quotes several series of investigations into Icukoplakia in which the rate of malignant change varies from 11 to 60 per cent. It is not so well known that lichen planus may also become malignant, but an increasing number of cases are being reported in the literature. Andrea~son” reported references to forty-six such previously recorded cases, and in eleven of them carcinoma had developed in plaquelike lesions. The rate of malignant change has been reported to be as high as 10 per cent in separate series by Dechaumelz and Warin. More recently,l” it has been stated t,hat t,he over-all malignancy rate in oral lichen planus is probably less than I per cent, but even at this rate the incidence is possibly greater than t,hc random chance assocG1tion of carcinoma with a relatively common disorder. In a discussion of lichen planus as long ago as 1931,1* emphasis was placed on the importance of differentiating the gross appearance of this condition from moniliasis. With the introduction of antibiotics, steroids, and cytotoxic drugs7 candidiasis of the mouth is a much more frequent occurrence, and it is now recognized that there exists a chronic hyperplastic formI in which thcl rvhite plaque cannot be removed by scraping. The histopathology in t,hcse cases is specific, but clinically the thick, edematous epithelial plaques are indistinguishable from “leukoplakia.” Recently, there has been a report of thirty wws oi candidial “leukoplakia” which were followetl for ‘L pears; in that time, nine underwent, malignant change.13 The patient whose case has been presented here was a nonsmoking, 1101~. syphilitic man with a dentally fit mouth; in the absence of mechanical, microbial, or fungal influences and, in view of the varying histology, it is impossible to account for the sudden appearance of the keratotic area observed in the mout-h or to state whether this represents only a variation of the lichen planus already present. Nevertheless, after consideration of the sinister clinical appearance and rapid growth of the lesion, and in view of the unpredictable occurrcncc 01’
854
Eggleston
Oral Surg. June, 1970
malignant conditions arising from apparently benign lesions of leukoplakia,ll total excision was adopted as the most satisfactory treatment in this case. The interest of this communication is that, although a consideration of the etiology has provided the grounds for a discussion point, it is the diagnosis at the clinical level which has formed the basis of treatment. Rather than subject the patient to repeated hospital admissions for periodic observation of the lesion, it was decided to remove the keratotic area completely and thus reduce to a minimum any risk to which the patient might have been exposed. SUMMARY A case of long-standing lichen planus in which a plaquelike lesion developed in the oral mucosa is reported. The significance of this lesion is discussed with regard to possible malignant change. Surgical removal is considered the most suitable form of treatment for all plaquelike lesions of the oral mucosa if they are of relatively small size. I wish to thank Professor Sir Robert Bradlaw for permission to use his cases and Mr. F. Nally for his encouragement and help with this paper. I would also like to thank the personnel of the Pathology Department and the Photographic Department of the Eastman Dental Hospital for their kind assistance and advice. REFERENCES 1. Andreason,
J.: Oral Lichen Planus, ORAL SURG. 25: 31, 1968. of Lichen Planus, Brit. Dent. J. 96: 1, 1954. 2. Cooke, B. E. D.: Oral Manifestations 3. Darling, A. I., and Crabb, H. 5. M.: Lichen Planus, ORAL Suaa. 7: 1276, 1954. Brocq, Q. J.: Lichen Plan des muqueuses et leuco plasies, Presse med. 27: 277-279, 1919. D., and Culver, G.: Lichen Planus of the Mouth Alone, Brit. J. Derm. 41: i: Montgomery, 45, 1929. 6. Wolf, J.: Lichen Planus and Leukoplakia, Arch. Derm. (Chicago) 63: 805, 1951. 7. Altman, J., and Perry, H.: Variations and Course of Lichen Planus, Arch. Derm. (Chicago) 84: 179-191, 1961. 8. Warin, R.: Epithelioma Following Lichen Planus in the Mouth, Brit. J. Derm. 72: 288, 1960. 9. Andreason, J., and Pindborg, J.: Canceruduiklug i oral lichen planus, Nord. Med. J. 70: 861-866, 1963. 10. Cooke, B. E. D.: Leukoplakia Buccalis and Oral Epithelial Naevi, Brit. J. Derm. 68: Xl174, 1956. 11. Silverman, S., Renstrup, G., and Pindborg, J.: Studies in Oral Leukoplakia, Acta Odont. Stand. 21: 271, 1963. 12. Dechaume et al.: Le lichen plan isole de la muquesse buceali, Presse med. 65: 2133-2135, 1957. 13. Association of Head and Neck Oncologists of Great Britain, Premalignant Lesions Affecting the Mouth, Pharynx and Larynx, Brit. Med. J. 2: 570, 1969. 14. Fox, H.: Lichen Planus Confined to the Mouth, Arch. Derm. (Chicago) 24: 1071-1082, 1931. 15. Lehner, T.: Oral Thrush in Acute Pseudo Membranous Candidiasis, ORAL SURG. 18: 27,
1964.