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Multiple fibroepithelial hyperplasias of the oral mucosa
Multiple fibroepithelial the oral mucosa A. Buchner, D.M.D., Tel Aviv, Israel SACKLER TEL
AVIV
SCHOOL
M.S.D.,”
OF MEDICINE
hyperplasias of and M. Sandbank, M.D.,** AND
SCHOOL
OF DENTAL
MEDICINE,
UNIVERSITY
Reported is a rare case of multiple fibroepitheliaf hyperplasias of the oral cavity, and the suggestion is made that several previous authors may have confused this lesion with focal epithelial hyperplasia (Heck’s disease). The differential diagnosis of multiple fibroepithelial hyperplasias is discussed in order to elucidate some of the confusions in the diagnosis of cases of multiple papules and nodules of the oral mucosa.
Several rare conditions
that affect the oral mucosa are manifested by multiple papules and nodules. In the past, these lesions were referred to as multiple papillomata,’ multiple polyps,* or multiple polypous hyperplasias .3 In 1965, Archard, Heck and Stanley4 described, in American Indians, an unusual hyperplastic reaction of the oral mucosa characterized clinically by discrete multiple papules. Since the main histologic feature was acanthosis of the epithelium, they designated this conditionfical epithelial hyperplasia. A review of the recent literature showed that, since the introduction of the termfocal epithelial hyperplasia, the name has been applied to a number of pathologic conditions which bear only superficial resemblance to that condition.” One of the lesions most confused with focal epithelial hyperplasia is an entity called multiple jibroepithelial hyperplasias. ,j This article describes a case of the rare condition multiple fibroepithelial hyperplasias and discusses the differential diagnosis in order to elucidate the confusion which exists when one faces the diagnosis of multiple papules and nodules in the oral cavity. CASE REPORT The patientwas a 62-year-old 12yearspreviously.
man who
had been born
in Russia,
and who
immigrated
to Israel
*Associate Professor, Department of Pathology, Sackler School of Medicine; and Head, Department of Oral Pathology and Oral Medicine, School of Dental Medicine, Tel Aviv University. **Senior Lecturer, Department of Dermatology, Sackler School of Medicine, Tel Aviv University, and Depart-
Volume 46 Number
Oral examination
Multiple jibroepithelial hyperplasias 35
Fig.
1. Lesions
Fig.
2. Multiple
on the buccal
lesions
mucosa.
on the tongue
revealed an edentelous mouth with multiple papules and nodules on the: lower
lip, upper lip, commissure, buccal mucosa, tongue, palate, and alveolar ridge of the manldib Ile
and
ma:ltilla (Figs. I - 3). The nodules were of various sizes and shapes (Fig. 4); some were petdunculate ,d and others were sessile. The surface of some lesions were pink in color and smooth ,w (hereas othl ers were white and corrugated. Although the patient had worn dentures for many years, he claimed that he had nfotic :ed the lesi ons only 5 years previously. There was no family history of similar lesions.
36
OralSurg.
Buchner and Sandbank
July, 1978
Fig.
Fig.
3. Lesions on the palate
4. Papules and nodules of various sizes and shapes
The general medical history revealed that the patient had been blind for many years because of glaucoma, and that he suffered from a peptic ulcer. No skin lesions were detected. The tentative diagnosis was focal epithelial hyperplasia (Heck’s disease). Several biopsy specimens taken from the lesions on the lip and buccal mucosa were subjected to light and electron microscopy. The specimens for light microscopy were fixed in IO per cent neutral formalin, and those for electron microscopy in 3 per cent cacodylate-buffered glutaraldehyde at pH 7.4 for 2 hours and postfixed for 2 hours in 2 per cent osmium tetroxide at pH 7.4. The tissue was embedded in Epon, and ultrathin sections were stained with many1 acetate and lead citrate.
Volume 46 Number 1
Mdtiplefibroepithelial hyperplasias 37
Fig. 5. Photomicrograph of a lesion exhibiting epithelial andfibroushyperplasia. (Magnification, x40.) light
microscopy
Stratifiedsquamous epitheliumwith a thin layer of parakeratinor orthokeratinwasfound. The epitheliumexhibited acanthosisand elongationof the rete ridges.A thick areaof hypetplastic connectivetissuewasseenbeneaththe epithelium.In certainareasthe connectivetissuewashighly cellular, whereasin othersit appearedto be composedmainly of densecollagenfibers(Fig. 5). Inflammatorycells, mainly mononuclear,were dispersedin the connectivetissue,and in certain areaspenetratedinto the epithelium.Sincebothepithelialandfibroushyperplasias werepresent,the histologicdiagnosiswasmultiplefibroepithelialhyperplasias,which ruled out the tentative diagnosisof focal epithelialhyperplasia. Electron
microscopy
No viral particlesweredetectedin the nucleior cytoplasmof theepithelialcells. However,the epithelialcells exhibitedan increasednumberof villous projectionsand foldingsof the plasma membrane.When one considersthe fact that the specimens were taken from the buccaland lip mucosa,the epithelialcellsshowedan increasednumberof the tonofilamentsaswell aspeculiar clumpingand whorlingof the tonofilaments(Fig. 6). DISCUSSION
From a review of the recent literature, we have the impression that various oral conditions which were manifested clinically as multiple papulesand nodules were diagnosedincorrectly as focal epithelial hyperplasia (Heck’s disease).j Focal epithelial hyperplasia is relatively common among Indians of the American continent,4 and in the Eskimo population of easternCanadaand western Greenland.‘, ’ It is rare in most other populations, and only single cases have been described among Caucasians.8 The characteristic histologic features of the lesions are acanthosisof the epithelium
38
OralSurg.
Buchner and Sandbank
Fii. 6. Electron x25.000.)
micrograph
showing
July,
peculiar
clumping
and whorling
of the tonofilaments.
1978
(Magnification,
with elongation and anastomosis of the rete ridges. 4 The underlying lamina propria is intact. Evaluation of histologic pictures presented in the literature as focal epithelial hyperplasia in certain cases in Caucasians, such as those of Waldman and Shelton9 or Anderson,” could be interpreted as multiple fibroepithelial hyperplasias since both epithelial and fibrous hyperplasia were present. Therefore, it seems that the condition of focal epithelial hyperplasia has been overdiagnosed, and we suggest that more attention be given to the differential diagnosis between focal epithelial hyperplasia and multiple fibroepithelial hyperplasias. The cause of multiple fibroepithelial hyperplasias is unknown, and probably because of the confusion in terminology, only a few cases have been described under this designation.” Several conditions with multiple lesions in the oral cavity have clinical features similar to those of multiple fibroepithelial hyperplasias and focal epithelial hyperplasia and should be considered in the differential diagnosis. Included in the differential list are the following: I. Multiple traumatic (irritation) fibromas due to local irritation. 2. Multiple verruca vulgaris. 3. Multiple oral papillomas, which may occur also in: a. Goltz syndrome (focal dermal hypoplasia syndrome). b. Cowden syndrome (multiple hamartoma and neoplasia syndrome). c. Acanthosis nigricans.
Volume 46
Number 1
MultipleJibroepithe2iul hyperplasius 39
4, Multiple oralneuromas inthesyndrome known asmultiple endocrine neoplasia,
type 3(multiple mucosal neuromas, pheochromocytoma, and medullary thyroid
carcinoma). 5. Multiple oral fibromas in tuberous sclerosis. The final diagnosis of all these oral lesions is based on histologic examination. A more detailed description of the individual lesions may be found in the textbook by Gorlin and associates.” We do not include in the differential list the term oral frorid pupillomatosis which is widely used in the dermatologic literature. 12,l3 The reason is that in most cases of oral florid papillomatosis the lesions are not scattered throughout the oral cavity,but rather are confined to the buccal mucosa and lips, and microscopically they exhibit the features outlined by Ackerman l4 for verrucous carcinoma. Thus, we agree with Eversole and SorensonlJ that recorded cases of oral florid papillomatosis are not actually examples of diffuse mucosal involvement by multiple benign papillomas but, rather, a separate wellrecognized entity-verrucous carcinoma, an aggressive, locally malignant neoplasm of the oral mucous membrane. REFERENCES I. Levy-Burg and Gerbay.: Un Cas de Papillomes de la Muqueuse Buccale, Ann. Mal. Vent%. 14: 608610, 1919. 2. Colman, R. S.: Multiple Polyps of the Buccal Mucous Membrane, ORAL SURG. 4: 466-468, 1951. 3. Bergenholtz, A.: Multiple Polypous Hyperplasias of the Oral Mucosa With Regression After Removal of Amalgam Fillings, Acta Odontol. Stand. 23: I I I - 134, 1965. 4. Archard, H. O., Heck, J. W., and Stanley, H. R.: Focal Epithelial Hyperplasia: An Unusual Oral Mucosal Lesion Found in Indian Children, ORAL SURG. 20: 201-212, 1965. 5. Gusterson, B. A., and Greenspan, J. S.: Multiple Polypoid Conditions of the Oral Mucosa, Br. J. Oral Surg. 12: 91-9.5, 1974. 6. Clausen, F. P., Mogeltofi, M., Roed-Petersen, B., and Pindborg, J. J.: Focal Epithelial Hyperplasia of the Oral Mucosa in a South-West Greenlandic Population, Stand. J. Dent. Res. 78: 287-294, 1970. 7. Jarvis, A., and Gorlin, R. J.: Focal Epithelial Hyperplasia in an Eskimo Population, ORAL SURG. 33: 227-228, 1972. 8. Bradman, P.: Focal Epithelial Hyperplasia (Heck’s disease), Br. J. Oral Surg. 8: 130-132, 1970. 9. Waldman, Cl. H., and Shelton, D. W.: Focal Epithelial Hyperplasia (Heck’s Disease) in an Adult Caucasian, ORAL SURG. 26: 124- 127, 1968. 10. Anderson, D. R.: Focal Epithelial Hyperplasia; Report of a Case in a South African Caucasoid, J. Dent. Assoc. S. Afr. 26: 32-35, 1971. I I. Gorlin, R. J., Pindborg, J. J., and Cohen, M. M.: Syndromes of the Head and Neck, ed. 2. New York, 1976, McGraw-Hill Book Co., Inc. 12. Wechsler, H. L., and Fisher, E. R.: Oral Florid Papillomatosis; Clinical, Pathological, and Electron Microscopic Observations, Arch. Dermatol. 86: 480-492, 1962. 13. Samitz, M. H., Ackerman, A. B., and Lantis, L. R.: Squamous Cell Carcinoma Arising at the Site of Oral Florid Papillomatosis, Arch. Dermatol. 96: 286-289, 1967. 14. Ackerman, L. V.: Verrucous Carcinoma of the Oral Cavity, Surgery 23: 670-678, 1948. 15. Eversole, L. R., and Sorenson, H. W.: Oral Florid Papillomatosis in Down’s Syndrome, ORAL SURF. 37: 202-207, 1974. Reprint requests to: Professor Amos Buchner School of Dental Medicine Tel Aviv University Ramat Aviv. Israel
AVIV
SCHOOL
M.S.D.,”
OF MEDICINE
hyperplasias of and M. Sandbank, M.D.,** AND
SCHOOL
OF DENTAL
MEDICINE,
UNIVERSITY
Reported is a rare case of multiple fibroepitheliaf hyperplasias of the oral cavity, and the suggestion is made that several previous authors may have confused this lesion with focal epithelial hyperplasia (Heck’s disease). The differential diagnosis of multiple fibroepithelial hyperplasias is discussed in order to elucidate some of the confusions in the diagnosis of cases of multiple papules and nodules of the oral mucosa.
Several rare conditions
that affect the oral mucosa are manifested by multiple papules and nodules. In the past, these lesions were referred to as multiple papillomata,’ multiple polyps,* or multiple polypous hyperplasias .3 In 1965, Archard, Heck and Stanley4 described, in American Indians, an unusual hyperplastic reaction of the oral mucosa characterized clinically by discrete multiple papules. Since the main histologic feature was acanthosis of the epithelium, they designated this conditionfical epithelial hyperplasia. A review of the recent literature showed that, since the introduction of the termfocal epithelial hyperplasia, the name has been applied to a number of pathologic conditions which bear only superficial resemblance to that condition.” One of the lesions most confused with focal epithelial hyperplasia is an entity called multiple jibroepithelial hyperplasias. ,j This article describes a case of the rare condition multiple fibroepithelial hyperplasias and discusses the differential diagnosis in order to elucidate the confusion which exists when one faces the diagnosis of multiple papules and nodules in the oral cavity. CASE REPORT The patientwas a 62-year-old 12yearspreviously.
man who
had been born
in Russia,
and who
immigrated
to Israel
*Associate Professor, Department of Pathology, Sackler School of Medicine; and Head, Department of Oral Pathology and Oral Medicine, School of Dental Medicine, Tel Aviv University. **Senior Lecturer, Department of Dermatology, Sackler School of Medicine, Tel Aviv University, and Depart-
Volume 46 Number
Oral examination
Multiple jibroepithelial hyperplasias 35
Fig.
1. Lesions
Fig.
2. Multiple
on the buccal
lesions
mucosa.
on the tongue
revealed an edentelous mouth with multiple papules and nodules on the: lower
lip, upper lip, commissure, buccal mucosa, tongue, palate, and alveolar ridge of the manldib Ile
and
ma:ltilla (Figs. I - 3). The nodules were of various sizes and shapes (Fig. 4); some were petdunculate ,d and others were sessile. The surface of some lesions were pink in color and smooth ,w (hereas othl ers were white and corrugated. Although the patient had worn dentures for many years, he claimed that he had nfotic :ed the lesi ons only 5 years previously. There was no family history of similar lesions.
36
OralSurg.
Buchner and Sandbank
July, 1978
Fig.
Fig.
3. Lesions on the palate
4. Papules and nodules of various sizes and shapes
The general medical history revealed that the patient had been blind for many years because of glaucoma, and that he suffered from a peptic ulcer. No skin lesions were detected. The tentative diagnosis was focal epithelial hyperplasia (Heck’s disease). Several biopsy specimens taken from the lesions on the lip and buccal mucosa were subjected to light and electron microscopy. The specimens for light microscopy were fixed in IO per cent neutral formalin, and those for electron microscopy in 3 per cent cacodylate-buffered glutaraldehyde at pH 7.4 for 2 hours and postfixed for 2 hours in 2 per cent osmium tetroxide at pH 7.4. The tissue was embedded in Epon, and ultrathin sections were stained with many1 acetate and lead citrate.
Volume 46 Number 1
Mdtiplefibroepithelial hyperplasias 37
Fig. 5. Photomicrograph of a lesion exhibiting epithelial andfibroushyperplasia. (Magnification, x40.) light
microscopy
Stratifiedsquamous epitheliumwith a thin layer of parakeratinor orthokeratinwasfound. The epitheliumexhibited acanthosisand elongationof the rete ridges.A thick areaof hypetplastic connectivetissuewasseenbeneaththe epithelium.In certainareasthe connectivetissuewashighly cellular, whereasin othersit appearedto be composedmainly of densecollagenfibers(Fig. 5). Inflammatorycells, mainly mononuclear,were dispersedin the connectivetissue,and in certain areaspenetratedinto the epithelium.Sincebothepithelialandfibroushyperplasias werepresent,the histologicdiagnosiswasmultiplefibroepithelialhyperplasias,which ruled out the tentative diagnosisof focal epithelialhyperplasia. Electron
microscopy
No viral particlesweredetectedin the nucleior cytoplasmof theepithelialcells. However,the epithelialcells exhibitedan increasednumberof villous projectionsand foldingsof the plasma membrane.When one considersthe fact that the specimens were taken from the buccaland lip mucosa,the epithelialcellsshowedan increasednumberof the tonofilamentsaswell aspeculiar clumpingand whorlingof the tonofilaments(Fig. 6). DISCUSSION
From a review of the recent literature, we have the impression that various oral conditions which were manifested clinically as multiple papulesand nodules were diagnosedincorrectly as focal epithelial hyperplasia (Heck’s disease).j Focal epithelial hyperplasia is relatively common among Indians of the American continent,4 and in the Eskimo population of easternCanadaand western Greenland.‘, ’ It is rare in most other populations, and only single cases have been described among Caucasians.8 The characteristic histologic features of the lesions are acanthosisof the epithelium
38
OralSurg.
Buchner and Sandbank
Fii. 6. Electron x25.000.)
micrograph
showing
July,
peculiar
clumping
and whorling
of the tonofilaments.
1978
(Magnification,
with elongation and anastomosis of the rete ridges. 4 The underlying lamina propria is intact. Evaluation of histologic pictures presented in the literature as focal epithelial hyperplasia in certain cases in Caucasians, such as those of Waldman and Shelton9 or Anderson,” could be interpreted as multiple fibroepithelial hyperplasias since both epithelial and fibrous hyperplasia were present. Therefore, it seems that the condition of focal epithelial hyperplasia has been overdiagnosed, and we suggest that more attention be given to the differential diagnosis between focal epithelial hyperplasia and multiple fibroepithelial hyperplasias. The cause of multiple fibroepithelial hyperplasias is unknown, and probably because of the confusion in terminology, only a few cases have been described under this designation.” Several conditions with multiple lesions in the oral cavity have clinical features similar to those of multiple fibroepithelial hyperplasias and focal epithelial hyperplasia and should be considered in the differential diagnosis. Included in the differential list are the following: I. Multiple traumatic (irritation) fibromas due to local irritation. 2. Multiple verruca vulgaris. 3. Multiple oral papillomas, which may occur also in: a. Goltz syndrome (focal dermal hypoplasia syndrome). b. Cowden syndrome (multiple hamartoma and neoplasia syndrome). c. Acanthosis nigricans.
Volume 46
Number 1
MultipleJibroepithe2iul hyperplasius 39
4, Multiple oralneuromas inthesyndrome known asmultiple endocrine neoplasia,
type 3(multiple mucosal neuromas, pheochromocytoma, and medullary thyroid
carcinoma). 5. Multiple oral fibromas in tuberous sclerosis. The final diagnosis of all these oral lesions is based on histologic examination. A more detailed description of the individual lesions may be found in the textbook by Gorlin and associates.” We do not include in the differential list the term oral frorid pupillomatosis which is widely used in the dermatologic literature. 12,l3 The reason is that in most cases of oral florid papillomatosis the lesions are not scattered throughout the oral cavity,but rather are confined to the buccal mucosa and lips, and microscopically they exhibit the features outlined by Ackerman l4 for verrucous carcinoma. Thus, we agree with Eversole and SorensonlJ that recorded cases of oral florid papillomatosis are not actually examples of diffuse mucosal involvement by multiple benign papillomas but, rather, a separate wellrecognized entity-verrucous carcinoma, an aggressive, locally malignant neoplasm of the oral mucous membrane. REFERENCES I. Levy-Burg and Gerbay.: Un Cas de Papillomes de la Muqueuse Buccale, Ann. Mal. Vent%. 14: 608610, 1919. 2. Colman, R. S.: Multiple Polyps of the Buccal Mucous Membrane, ORAL SURG. 4: 466-468, 1951. 3. Bergenholtz, A.: Multiple Polypous Hyperplasias of the Oral Mucosa With Regression After Removal of Amalgam Fillings, Acta Odontol. Stand. 23: I I I - 134, 1965. 4. Archard, H. O., Heck, J. W., and Stanley, H. R.: Focal Epithelial Hyperplasia: An Unusual Oral Mucosal Lesion Found in Indian Children, ORAL SURG. 20: 201-212, 1965. 5. Gusterson, B. A., and Greenspan, J. S.: Multiple Polypoid Conditions of the Oral Mucosa, Br. J. Oral Surg. 12: 91-9.5, 1974. 6. Clausen, F. P., Mogeltofi, M., Roed-Petersen, B., and Pindborg, J. J.: Focal Epithelial Hyperplasia of the Oral Mucosa in a South-West Greenlandic Population, Stand. J. Dent. Res. 78: 287-294, 1970. 7. Jarvis, A., and Gorlin, R. J.: Focal Epithelial Hyperplasia in an Eskimo Population, ORAL SURG. 33: 227-228, 1972. 8. Bradman, P.: Focal Epithelial Hyperplasia (Heck’s disease), Br. J. Oral Surg. 8: 130-132, 1970. 9. Waldman, Cl. H., and Shelton, D. W.: Focal Epithelial Hyperplasia (Heck’s Disease) in an Adult Caucasian, ORAL SURG. 26: 124- 127, 1968. 10. Anderson, D. R.: Focal Epithelial Hyperplasia; Report of a Case in a South African Caucasoid, J. Dent. Assoc. S. Afr. 26: 32-35, 1971. I I. Gorlin, R. J., Pindborg, J. J., and Cohen, M. M.: Syndromes of the Head and Neck, ed. 2. New York, 1976, McGraw-Hill Book Co., Inc. 12. Wechsler, H. L., and Fisher, E. R.: Oral Florid Papillomatosis; Clinical, Pathological, and Electron Microscopic Observations, Arch. Dermatol. 86: 480-492, 1962. 13. Samitz, M. H., Ackerman, A. B., and Lantis, L. R.: Squamous Cell Carcinoma Arising at the Site of Oral Florid Papillomatosis, Arch. Dermatol. 96: 286-289, 1967. 14. Ackerman, L. V.: Verrucous Carcinoma of the Oral Cavity, Surgery 23: 670-678, 1948. 15. Eversole, L. R., and Sorenson, H. W.: Oral Florid Papillomatosis in Down’s Syndrome, ORAL SURF. 37: 202-207, 1974. Reprint requests to: Professor Amos Buchner School of Dental Medicine Tel Aviv University Ramat Aviv. Israel