Journal of the American Academy of Dermatology
Sayre and Agin 19. Sayre RM, Agin PP, LeVee GJ, et al. A comparison of in vivo and in vitro testing of sunscreening products. Photochern Photobiol J 979;29:559-66. 20. Sayre RM, Agin PP, Desrochers DL, et al. Sunscreen testing methods: in vitro predictions of effectiveness. J Soc Cosmet Chem 1980;31:133-43.
21. Brown S, Ditfey BL. The effect of applied thickness on sunscreen protection: in vivo and in vitro studies. Photochem Photobiol 1986;44:509-13. 22. Groves GA, Agin PP, Sayre RM. In vitro and in vivo methods to define sunscreen protection. Aust J Dermatol 1979;20:112-9.
Pigmented Bowen's disease arising from pigmented seborrheic keratoses Stephanie F. Marschall, MD,a Salve G. Ronan, MD,b and Mary C. Massa, MD c
Chicago and Maywood, Illinois We report three cases of pigmented Bowen's disease that clinically and histologically had features of seborrheic keratoses. We speculateabout the mechanism of pigmentation inthese lesionsand suggest that they arise from pigmented seborrheickeratoses. (J AM ACAD DERMATOl 1990;23:440-4.) Bowen's disease is an intraepidermal carcinoma that most commonly presents as an asymptomatic, scaly, erythematous plaque on the trunk or extremities. 1 Reports of pigmented lesions of Bowen's disease are rare. In 1970 Lloyd- first reported a case of multicentric pigmented Bowen's disease of the groin. Since then, additional cases of pigmented Bowen's disease of the anogenital region have been published. 3,4 Pigmented Bowen's disease outside the anogenital area has been rarely reported. Two occurred in the interdigital web spaces.f- 6 and two presented as solitary pigmented lesions, one on the neck? and an- Fig. 1. Case 1. Verrucous pigmented plaque on right other on the leg," Ragi et a1. 6 reviewed the records forearm. Central hypopigmented area represents previof 420 cases of Bowen's disease and found seven ous biopsy site. ( 1.7%) to be pigmented, none of which involved the anogenital area. Seborrheic keratoses are benign epidermal neo- scribes three cases of pigmented Bowen's disease plasms that are commonly pigmented. Formation of outside the anogenital area. in situ carcinoma or Bowen's disease within a seborCASE REPORTS rheic keratosis is seen rarely.v!! This report de-
Case 1
From the Departments of Dermatology" and Pathology.P University of Illinois at Chicago, and the Section of Dermatology, Department of Medicine, Stritch School of Medicine, Maywood." Accepted for publication Nov. 16, 1989. Reprint requests: Stephanie F. Marschall, MD, Dcpartrncn t of Dermatology, University of Illinois at Chicago, 808 S. Wood St., CME 376, Chicago, IL 60612.
16/1/18300
440
A 65-year-old white man had had a pigmented lesion on the left arm for many years. Examination revealed an irregular, 2.0 em, well-defined, brown, verrucous plaque on the extensorsurface of the left arm (Fig. 1).The lesion was histologically examined and subsequently excised. Histologic sections revealed papillomatosis, marked hyperkeratosis, parakeratosis, and acanthosis. Areas of mature keratinocytes without cellular atypia were seen
Volume 23 Number 3, Part 1 September 1990
Pigmented Bowen's disease 441
Fig. 2. Case 1. Section of keratotic lesion with areas of mature keratinocytes without cellular atypia, adjacent to foci of immature cells. (Hematoxylin-eosin stain; X5.)
. Fig. 3. Case 1. Section of keratotic lesion showing cellular atypia and increased mitotic figures. (Hematoxylin-eosin stain; XlOO.)
adjacent to foci of immature cells (Fig. 2). Pleomorphic nuclei and increased mitoses in the stratum malpighii with numerous dyskeratotic cells were seen arranged in a haphazard fashion (Fig. 3). Fontana's stain for melanin showed increased pigmentation of the keratinocytes, most prominent in the basal cell layer. This was seen in a uniform pattern in the area of the seborrheic keratosis, but more haphazardly distributed in the neoplastic areas. The superficial dermis showed a lymphocytic infiltrate and melanophages. No invasion was seen.
Histologic sections showed marked hyperkeratosis and papillomatosis, with pseudohorn cysts and scattered areas of acanthosis, adjacent to foci of immature large cells with pleomorphic nuclei. Numerous mitotic figures and dyskeratotic cells were seen dispersed within and adjacent to areas of normal cellular maturation. Increased melanin was seen both with hematoxylin and eosin stain (Fig. 4) and with Fontana stain. The distribution of melanin was similar to that of case I. In the papillary dermis were aggregates of melanophages (Fig. 5).
Case 2
Case 3
A 46-year-old black man had had a pigmented lesion on the right arm for 3 years. Examination revealed a 2.0 ern, dark brown, verrucous plaque with irregular borders. The entire lesion was excised.
A 60-year-old white man had had a pigmented lesion on the right flank for many years. Examination revealed an irregular, 4 X 3 em, dark brown plaque. The entire lesion was excised.
442
Journal of the American Acad emy of Dermatology
Marschall et al.
'.... ' ."
::. ~.".:'b "". ~
"'"',
.
Fig. 4. Case 2. Neoplastic keratinocytes showing melanin pigment in cytoplasm. (Hematoxylin-eosin stain; X200.)
Fig. 5. Case 2. Aggregates of melanophages in dermis. (Hematoxylin-eosin stain; X200.)
Histologic sections revealed marked hyperkeratosis, papillomatosis, and acanthosis, with pseudohorn cysts in the epidermis, adjacent to areas with features of Bowen's disease (Fig. 6). In the acanthotic epidermis were areas of cellular disarray with loss of the normal maturation pattern. The keratinocytes contained pleomorphic nuclei. with prominent nucleoli. Many mitotic figures were seen, and some cells were binuclear. Increased melanin was seen within the neoplastic keratinocytes as well as in the area of seborrheic keratosis. Fontana's stain showed increased melanin distributed as in cases 1 and 2. In addition, a number of highly dendritic melanocytes were seen both in the neoplastic areas and in the area of the seborrheic keratosis but more prominently in the former .
In the papillary dermis was a dense, lymphocytic infiltrate with a few melanophages.
DISCUSSION
To our knowledge this is only the third report of pigmented Bowen's disease outside an intertriginous area. Furthermore, it appears from histologic examination of our cases that pigmented lesions of Bowen's disease may arise in a preexisting seborrheic keratosis as was seen in Bloch's case.s In some of the cases reviewed by Ragi et a1. 6 and in the case reported by Scarborough et al.," pigmented Bowen's disease may also have arisen in a seborrheic kerato-
Volume 23 Number 3, Part 1 September 1990
Pigmented Bowen's disease 443
Fig. 6. Case 3. Low-power view of seborrheic keratoses with changes of Bowen's disease within. (Hematoxylin-eosin stain; XS.)
sis and further sectioning of the specimens may have revealed the transformation. The mechanism responsible for pigmentation in epidermal neoplasms is not always clear. Burgoyne et a1. 12 demonstrated that melanocytic hyperplasia and increased melanin synthesis occurs in experimentally induced epidermal cell hyperplasia. Pinkus et a1. 13 emphasized the interrelationship between melanocytes and keratinocytes under physiologic conditions. However, in neoplasms the amount of pigment may vary but does not influence the degree of malignancy." Ragi et a1. did not reach any conclusions on the cause of the pigmentation in their cases. Our cases showed increased pigment within the neoplastic keratinocytes and melanophages in the papillary dermis. We speculate that the pigmentation noted clinically is due to increased melanin synthesis with an increase in uptake by keratinocytes, an increased number of melanocytes, and the presence of dermal melanophages. Two thirds of seborrheic keratoses are shown to be pigmented when silver staining is done. 14 Becker 15 found that pigmentation in the basal layer occurs first and is followed by the more superficial layers. The melanocytic hyperplasia seen with epidermal hyperplasia, and the interaction between these two populations of cells accounts for the pigmentation usually seen in seborrheic keratoses. Given this phenomenon, it is not surprising that Bowen's disease arising from a preexisting seborrheic keratosis is pigmented.
Bowenoid transformation of seborrheic keratoses has been infrequently reported.P'! Christeler and Delacretaz? reported an axillary lesion that also demonstrated glandular metaplasia. Rahbari'" reviewed 3000 cases and found seven (0.2%) with changes of Bowen's disease located in the center of a typical benign seborrheic keratosis. Bloch8 stressed the importance of demonstrating a transition zone between the two lesions as proof of the transformation. Kwitten 11 described 19 cases of malignant change in seborrheic keratoses that ranged from inconspicuous microscopic foci to features indistinguishable from Bowen's disease. The areas of intraepidermal carcinoma seen in our patients were not simply small foci of bowenoid changes but true Bowen's disease. We cannot be certain that the hyperkeratosis and acanthosis in the areas interpreted as benign seborrheic keratoses in our cases were not the result of Bowen's disease. This is unlikely because no abnormal cell maturation or atypia was seen in these areas. Pigmented Bowen's disease may possibly occur de novo, and the pigmentation may be due to melanocytic hyperplasia. Too few reports exist in the literature to reach any conclusions on the incidence of de novo pigmented Bowen's disease. REFERENCES I. Lever WF, Schaumburg-Lever G. Tumors and cysts ofthc epidermis. In: Histopathology of the skin. 6th ed. Philadelphia: JB Lippincott, 1983:496-8. 2. Lloyd KM. Multicentric pigmented Bowen's disease of the groin. Arch Derma tal 1970; I01:48-51.
Journal of the American Academy of Dermatology
Marschall et al. 3. Wilson HT. Multicentric pigmented Bowen's disease. Br J Dermatol 1971 ;84: 183-4. 4. Wagner RF, GrandeDJ. Solitarypigmented Bowen'sdisease of the scrotum. J Dermatol Surg Oncol 1986;12: II 14-5. 5. Burns DA. Bilateral pigmented Bowen's disease of the webspaces of the feet. Clin Exp Dermatol 1986;6:435-7. 6. Ragi G, Turner MS, KleinLE, et al. Pigmented Bowen's disease and review of 420 Bowen's disease lesions. J Dermatol Surg Oncol 1988;14:765-9. 7. Scarborough DA, Bisaccia EP, Yoder FW. Solitary pigmented Bowen's disease. Arch Dermatol 1982;118:954-5. 8. Bloch PH. Transformation ofseborrheic keratosis into Bowen's disease. J Cutan Pathol 1978;5:361-7. 9. Christeler PA, Delacretaz J. Verrues seborrneiques ilt transformation maligne. Dermatologica 1966;133:33-9.
10. Rahbari H. Bowenoid transformation of seborrheic verrucae (keratoses). Br J DermatoI1979;101:459-63. 11. Kwitten 1. Malignant changesin seborrheic keratoses. Mt Sinai J Med (NY) 1974;41:792-801. 12. Burgoyne PH, Heston WE, Hartwell JL, et al. Cutaneous melanin production in mice following application of the carcinogen 5,9, IO-trimethoyl-l ,2-benzanthracene. JNCI 1949;10:665-88. 13. Pinkus H, Staricco RJ, Kropp PJ, et al, The symbiosis of rnelanocytes and human epidermis under normal and abnormalconditions. In Gordon M, ed. Pigment cell biology. New York: Academic Press, 1959. 14. LennoxB.Pigment patterns in epithelial tumors of the skin. J Pathol Bacterol 1949;61:587-98. 15. Becker SW. Melanotic neoplasms of the skin. Am J Cancer 1934;22:17-40.
Cheilitis granulomatosa: Report of six cases and review of the literature Carl M. Allen, DDS, MSD,a Charles Carnisa, MD,b Sahar Hamzeh, DDS,a and Linda Stephens, BSC Columbus and Cleveland, Ohio, and Los Angeles, California Six cases ofcheilitis granulomatosa, a rare inflammatory disorder ofunknown origin, are re-
ported. Thecondition produces nontender, persistent swelling of one or both lips and affects primarily young adults. Histologically, nonnecrotizing granulomatous inflammation is seen. Theclinical findings and results oftherapyin these sixcases are presented. Onepatientwas treated with hydroxychloroquine sulfate (Plaquenil) that stabilized the process. One of our patients hadvesicular-appearing lesions. Microscopic examination showed the lesions to be dilated superficial lymphatic channels, a finding that to our knowledge has not been previously described. (1 AM ACAD DERMATOL 1990;23:444-50.) Cheilitis granulomatosa (CG) is a rare inflammatory disorder of unknown origin that was described by Miescher! in 1945. CG usually affects young adults and is characterized clinicallyby diffuse, nontender,soft to firm swelling of one or both Iips.? Histologically, nonnecrotizing granulomas are seen, as well as edema, lymphangiectasia, and perivascular lymphocytic infiltration. A variety of treatments havebeenusedbut none has been proved From theCollege ofDentistry, Section ofDiagnosticServices, TheOhio State University, Columbus," theDepartment ofDermatology, The Cleveland Clinic Foundation," andtheCollege ofMedicine, UniversityofSouthern California," Accepted for publication Dec. 16,1989. Reprint requests: Carl M. Allen, DDS, 305 W. 12th Ave., Columbus, OH 43210. 16/1/19017
444
to be uniformly effective.' Several authors have suggested that CO or a CO-like condition may be associated with Melkersson-Rosenthal syndrome or sarcoidosis."? The purpose of this article is to describe six patients with CG and to review the literature. CASE REPORTS (Table I)
Case 1 A 36-year-old white manhad persistent swelling of the upperandlower lip for6 years. Previous therapywithantihistamines and intralesional injections of triamcinolone acetonide was unsuccessful. Nosystemic signs ofregional enteritis or sarcoidosis were present. Physical examination showed an enlarged, soft, nontenderlower lip and a slightly swollen upper lip. No intraorallesions were apparent. Tissue sampled from the
Sayre and Agin 19. Sayre RM, Agin PP, LeVee GJ, et al. A comparison of in vivo and in vitro testing of sunscreening products. Photochern Photobiol J 979;29:559-66. 20. Sayre RM, Agin PP, Desrochers DL, et al. Sunscreen testing methods: in vitro predictions of effectiveness. J Soc Cosmet Chem 1980;31:133-43.
21. Brown S, Ditfey BL. The effect of applied thickness on sunscreen protection: in vivo and in vitro studies. Photochem Photobiol 1986;44:509-13. 22. Groves GA, Agin PP, Sayre RM. In vitro and in vivo methods to define sunscreen protection. Aust J Dermatol 1979;20:112-9.
Pigmented Bowen's disease arising from pigmented seborrheic keratoses Stephanie F. Marschall, MD,a Salve G. Ronan, MD,b and Mary C. Massa, MD c
Chicago and Maywood, Illinois We report three cases of pigmented Bowen's disease that clinically and histologically had features of seborrheic keratoses. We speculateabout the mechanism of pigmentation inthese lesionsand suggest that they arise from pigmented seborrheickeratoses. (J AM ACAD DERMATOl 1990;23:440-4.) Bowen's disease is an intraepidermal carcinoma that most commonly presents as an asymptomatic, scaly, erythematous plaque on the trunk or extremities. 1 Reports of pigmented lesions of Bowen's disease are rare. In 1970 Lloyd- first reported a case of multicentric pigmented Bowen's disease of the groin. Since then, additional cases of pigmented Bowen's disease of the anogenital region have been published. 3,4 Pigmented Bowen's disease outside the anogenital area has been rarely reported. Two occurred in the interdigital web spaces.f- 6 and two presented as solitary pigmented lesions, one on the neck? and an- Fig. 1. Case 1. Verrucous pigmented plaque on right other on the leg," Ragi et a1. 6 reviewed the records forearm. Central hypopigmented area represents previof 420 cases of Bowen's disease and found seven ous biopsy site. ( 1.7%) to be pigmented, none of which involved the anogenital area. Seborrheic keratoses are benign epidermal neo- scribes three cases of pigmented Bowen's disease plasms that are commonly pigmented. Formation of outside the anogenital area. in situ carcinoma or Bowen's disease within a seborCASE REPORTS rheic keratosis is seen rarely.v!! This report de-
Case 1
From the Departments of Dermatology" and Pathology.P University of Illinois at Chicago, and the Section of Dermatology, Department of Medicine, Stritch School of Medicine, Maywood." Accepted for publication Nov. 16, 1989. Reprint requests: Stephanie F. Marschall, MD, Dcpartrncn t of Dermatology, University of Illinois at Chicago, 808 S. Wood St., CME 376, Chicago, IL 60612.
16/1/18300
440
A 65-year-old white man had had a pigmented lesion on the left arm for many years. Examination revealed an irregular, 2.0 em, well-defined, brown, verrucous plaque on the extensorsurface of the left arm (Fig. 1).The lesion was histologically examined and subsequently excised. Histologic sections revealed papillomatosis, marked hyperkeratosis, parakeratosis, and acanthosis. Areas of mature keratinocytes without cellular atypia were seen
Volume 23 Number 3, Part 1 September 1990
Pigmented Bowen's disease 441
Fig. 2. Case 1. Section of keratotic lesion with areas of mature keratinocytes without cellular atypia, adjacent to foci of immature cells. (Hematoxylin-eosin stain; X5.)
. Fig. 3. Case 1. Section of keratotic lesion showing cellular atypia and increased mitotic figures. (Hematoxylin-eosin stain; XlOO.)
adjacent to foci of immature cells (Fig. 2). Pleomorphic nuclei and increased mitoses in the stratum malpighii with numerous dyskeratotic cells were seen arranged in a haphazard fashion (Fig. 3). Fontana's stain for melanin showed increased pigmentation of the keratinocytes, most prominent in the basal cell layer. This was seen in a uniform pattern in the area of the seborrheic keratosis, but more haphazardly distributed in the neoplastic areas. The superficial dermis showed a lymphocytic infiltrate and melanophages. No invasion was seen.
Histologic sections showed marked hyperkeratosis and papillomatosis, with pseudohorn cysts and scattered areas of acanthosis, adjacent to foci of immature large cells with pleomorphic nuclei. Numerous mitotic figures and dyskeratotic cells were seen dispersed within and adjacent to areas of normal cellular maturation. Increased melanin was seen both with hematoxylin and eosin stain (Fig. 4) and with Fontana stain. The distribution of melanin was similar to that of case I. In the papillary dermis were aggregates of melanophages (Fig. 5).
Case 2
Case 3
A 46-year-old black man had had a pigmented lesion on the right arm for 3 years. Examination revealed a 2.0 ern, dark brown, verrucous plaque with irregular borders. The entire lesion was excised.
A 60-year-old white man had had a pigmented lesion on the right flank for many years. Examination revealed an irregular, 4 X 3 em, dark brown plaque. The entire lesion was excised.
442
Journal of the American Acad emy of Dermatology
Marschall et al.
'.... ' ."
::. ~.".:'b "". ~
"'"',
.
Fig. 4. Case 2. Neoplastic keratinocytes showing melanin pigment in cytoplasm. (Hematoxylin-eosin stain; X200.)
Fig. 5. Case 2. Aggregates of melanophages in dermis. (Hematoxylin-eosin stain; X200.)
Histologic sections revealed marked hyperkeratosis, papillomatosis, and acanthosis, with pseudohorn cysts in the epidermis, adjacent to areas with features of Bowen's disease (Fig. 6). In the acanthotic epidermis were areas of cellular disarray with loss of the normal maturation pattern. The keratinocytes contained pleomorphic nuclei. with prominent nucleoli. Many mitotic figures were seen, and some cells were binuclear. Increased melanin was seen within the neoplastic keratinocytes as well as in the area of seborrheic keratosis. Fontana's stain showed increased melanin distributed as in cases 1 and 2. In addition, a number of highly dendritic melanocytes were seen both in the neoplastic areas and in the area of the seborrheic keratosis but more prominently in the former .
In the papillary dermis was a dense, lymphocytic infiltrate with a few melanophages.
DISCUSSION
To our knowledge this is only the third report of pigmented Bowen's disease outside an intertriginous area. Furthermore, it appears from histologic examination of our cases that pigmented lesions of Bowen's disease may arise in a preexisting seborrheic keratosis as was seen in Bloch's case.s In some of the cases reviewed by Ragi et a1. 6 and in the case reported by Scarborough et al.," pigmented Bowen's disease may also have arisen in a seborrheic kerato-
Volume 23 Number 3, Part 1 September 1990
Pigmented Bowen's disease 443
Fig. 6. Case 3. Low-power view of seborrheic keratoses with changes of Bowen's disease within. (Hematoxylin-eosin stain; XS.)
sis and further sectioning of the specimens may have revealed the transformation. The mechanism responsible for pigmentation in epidermal neoplasms is not always clear. Burgoyne et a1. 12 demonstrated that melanocytic hyperplasia and increased melanin synthesis occurs in experimentally induced epidermal cell hyperplasia. Pinkus et a1. 13 emphasized the interrelationship between melanocytes and keratinocytes under physiologic conditions. However, in neoplasms the amount of pigment may vary but does not influence the degree of malignancy." Ragi et a1. did not reach any conclusions on the cause of the pigmentation in their cases. Our cases showed increased pigment within the neoplastic keratinocytes and melanophages in the papillary dermis. We speculate that the pigmentation noted clinically is due to increased melanin synthesis with an increase in uptake by keratinocytes, an increased number of melanocytes, and the presence of dermal melanophages. Two thirds of seborrheic keratoses are shown to be pigmented when silver staining is done. 14 Becker 15 found that pigmentation in the basal layer occurs first and is followed by the more superficial layers. The melanocytic hyperplasia seen with epidermal hyperplasia, and the interaction between these two populations of cells accounts for the pigmentation usually seen in seborrheic keratoses. Given this phenomenon, it is not surprising that Bowen's disease arising from a preexisting seborrheic keratosis is pigmented.
Bowenoid transformation of seborrheic keratoses has been infrequently reported.P'! Christeler and Delacretaz? reported an axillary lesion that also demonstrated glandular metaplasia. Rahbari'" reviewed 3000 cases and found seven (0.2%) with changes of Bowen's disease located in the center of a typical benign seborrheic keratosis. Bloch8 stressed the importance of demonstrating a transition zone between the two lesions as proof of the transformation. Kwitten 11 described 19 cases of malignant change in seborrheic keratoses that ranged from inconspicuous microscopic foci to features indistinguishable from Bowen's disease. The areas of intraepidermal carcinoma seen in our patients were not simply small foci of bowenoid changes but true Bowen's disease. We cannot be certain that the hyperkeratosis and acanthosis in the areas interpreted as benign seborrheic keratoses in our cases were not the result of Bowen's disease. This is unlikely because no abnormal cell maturation or atypia was seen in these areas. Pigmented Bowen's disease may possibly occur de novo, and the pigmentation may be due to melanocytic hyperplasia. Too few reports exist in the literature to reach any conclusions on the incidence of de novo pigmented Bowen's disease. REFERENCES I. Lever WF, Schaumburg-Lever G. Tumors and cysts ofthc epidermis. In: Histopathology of the skin. 6th ed. Philadelphia: JB Lippincott, 1983:496-8. 2. Lloyd KM. Multicentric pigmented Bowen's disease of the groin. Arch Derma tal 1970; I01:48-51.
Journal of the American Academy of Dermatology
Marschall et al. 3. Wilson HT. Multicentric pigmented Bowen's disease. Br J Dermatol 1971 ;84: 183-4. 4. Wagner RF, GrandeDJ. Solitarypigmented Bowen'sdisease of the scrotum. J Dermatol Surg Oncol 1986;12: II 14-5. 5. Burns DA. Bilateral pigmented Bowen's disease of the webspaces of the feet. Clin Exp Dermatol 1986;6:435-7. 6. Ragi G, Turner MS, KleinLE, et al. Pigmented Bowen's disease and review of 420 Bowen's disease lesions. J Dermatol Surg Oncol 1988;14:765-9. 7. Scarborough DA, Bisaccia EP, Yoder FW. Solitary pigmented Bowen's disease. Arch Dermatol 1982;118:954-5. 8. Bloch PH. Transformation ofseborrheic keratosis into Bowen's disease. J Cutan Pathol 1978;5:361-7. 9. Christeler PA, Delacretaz J. Verrues seborrneiques ilt transformation maligne. Dermatologica 1966;133:33-9.
10. Rahbari H. Bowenoid transformation of seborrheic verrucae (keratoses). Br J DermatoI1979;101:459-63. 11. Kwitten 1. Malignant changesin seborrheic keratoses. Mt Sinai J Med (NY) 1974;41:792-801. 12. Burgoyne PH, Heston WE, Hartwell JL, et al. Cutaneous melanin production in mice following application of the carcinogen 5,9, IO-trimethoyl-l ,2-benzanthracene. JNCI 1949;10:665-88. 13. Pinkus H, Staricco RJ, Kropp PJ, et al, The symbiosis of rnelanocytes and human epidermis under normal and abnormalconditions. In Gordon M, ed. Pigment cell biology. New York: Academic Press, 1959. 14. LennoxB.Pigment patterns in epithelial tumors of the skin. J Pathol Bacterol 1949;61:587-98. 15. Becker SW. Melanotic neoplasms of the skin. Am J Cancer 1934;22:17-40.
Cheilitis granulomatosa: Report of six cases and review of the literature Carl M. Allen, DDS, MSD,a Charles Carnisa, MD,b Sahar Hamzeh, DDS,a and Linda Stephens, BSC Columbus and Cleveland, Ohio, and Los Angeles, California Six cases ofcheilitis granulomatosa, a rare inflammatory disorder ofunknown origin, are re-
ported. Thecondition produces nontender, persistent swelling of one or both lips and affects primarily young adults. Histologically, nonnecrotizing granulomatous inflammation is seen. Theclinical findings and results oftherapyin these sixcases are presented. Onepatientwas treated with hydroxychloroquine sulfate (Plaquenil) that stabilized the process. One of our patients hadvesicular-appearing lesions. Microscopic examination showed the lesions to be dilated superficial lymphatic channels, a finding that to our knowledge has not been previously described. (1 AM ACAD DERMATOL 1990;23:444-50.) Cheilitis granulomatosa (CG) is a rare inflammatory disorder of unknown origin that was described by Miescher! in 1945. CG usually affects young adults and is characterized clinicallyby diffuse, nontender,soft to firm swelling of one or both Iips.? Histologically, nonnecrotizing granulomas are seen, as well as edema, lymphangiectasia, and perivascular lymphocytic infiltration. A variety of treatments havebeenusedbut none has been proved From theCollege ofDentistry, Section ofDiagnosticServices, TheOhio State University, Columbus," theDepartment ofDermatology, The Cleveland Clinic Foundation," andtheCollege ofMedicine, UniversityofSouthern California," Accepted for publication Dec. 16,1989. Reprint requests: Carl M. Allen, DDS, 305 W. 12th Ave., Columbus, OH 43210. 16/1/19017
444
to be uniformly effective.' Several authors have suggested that CO or a CO-like condition may be associated with Melkersson-Rosenthal syndrome or sarcoidosis."? The purpose of this article is to describe six patients with CG and to review the literature. CASE REPORTS (Table I)
Case 1 A 36-year-old white manhad persistent swelling of the upperandlower lip for6 years. Previous therapywithantihistamines and intralesional injections of triamcinolone acetonide was unsuccessful. Nosystemic signs ofregional enteritis or sarcoidosis were present. Physical examination showed an enlarged, soft, nontenderlower lip and a slightly swollen upper lip. No intraorallesions were apparent. Tissue sampled from the
