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Balloon cell transformation in multiple dysplastic nevi
290
Journal of the American Academy of Dermatology
Brief communications
ative, associated scalp lesions may be seen in 30% of lupus erythematosus profundus cases.6 Periorbital and eyelid edema resulting from lupus erythematosus profundus has been reported in the ophthalmologic literature. In one report,? persistent unilateral eyelid edema was the first recognized sign of lupus erythematosus profundus in three patients, and one case was associated with proptosis. These patients' symptoms improved in response to antimalarial therapy, and all had lesions of discoid LE, which were not present in our patient. One other report described unilateral proptosis in a case of systemic LE with associated swelling of the periorbital tissues. s An explanation for the proptosis was not given, although the clinical description was consistent with that of lupus erythematosus profundus. Another case of unilateral periorbital edema associated with ipsilateral lupus erythematosus profundus of the cheek has been reported in the dental literature. In this case, the trauma of oral surgery possibly exacerbated the panniculitis, and it was postulated that lymphatic obstruction, rather than actual involvement of the periorbital fat, had caused the periorbital edema. Although trauma apparently was not a contributing factor in our case, it has preceded other cases oflupus erythematosus profundus. I. 2, 10 In addition to the ocular findings in our patient, two other aspects of the case are worthy of comment. Initial lesions of lupus erythematosus profundus are usually freely movable. I , \0 Our patient's facial nodule on the left side was so indurated and fixed that we were concerned about the possibility of a neoplasm, although we were aware ofthe patient's past history oflupus erythematosus profundus. In addition, our patient had a long-standing nonscarring patchy hair loss that was clinically consistent with alopecia areata, which progressed despite treatment with antimalarial agents. This emphasizes that normalappearing skin with no clinically detectable depressions or indurations may overlie lesions of lupus erythematosus profundus. REFERENCES 1. Izumi AK, Takiguchi P. Lupus erythematosus panniculitis. Arch Dermatol 1983;119:61-4. 2. Tuffanelli DL. Lupus erythematosus (panniculitis) profundus: a classic revisited--eommentary and report of22 cases. Hawaii Med J 1982;41:394-7. 3. Zweiman B, Tomar RH, Gross PRo Lupus erythematosus profundus following thrombocytopenic purpura. Arch Dermatol 1975;111 :347-51. 4. Winkelmann RK. Panniculitis in connective tissue disease. Arch Dermatol1983;ll9:336-44. 5. Diaz-Jouanen E, DeHoratius RJ, Alarcon-Segovia D, et al. Systemic lupus erythematosus presenting as panniculitis (lupus profundus). Ann Intern Med 1975;82:376-9. 6. Sanchez NP, Peters MS, Winkelmann RK. The histopathology of lupus erythematosus panniculitis. J AM ACAD DERMATOL 1981;5:673-80. 7. Nowinski T, Bernardino V, Naidoff M, et al. Ocular involvement in lupus erythematosus profundus (panniculitis). Ophthalmology 1982;89:1149-54.
8. Brenner EH, Shock JP. Proptosis secondary to systemic lupus erythematosus. Arch OphthalmoI1974;91:81-2. 9. Klein SA, Tomaro AJ. Lupus erythematosus profundus: review of the literature and report of case. 1 Oral Surg 1975;33:454-7. 10. Izumi AK. Lupus erythematosus panniculitis. Clin DermatoI1985;3:69-77.
Balloon cell transformation in multiple dysplastic nevi Bruce R. Smoller, MD,a.b Susan Kindel, MD,b N. Scott McNutt, MD,a.b Mark H. Gray,a and Amy Hsua New York. New York Melanocytic nevi composed entirely of balloon cells are rare, I as are balloon cell melanomas? Balloon cells are melanocytes with extensive, pale-staining, and vacuolated cytoplasm. This histologic pattern is thought to be caused by a defect in melanosome formation that leads to faulty melanin production. 3 Although focal ballooning transformation is not uncommon, extensive change has not been reported in a dysplastic nevus. We recently saw two patients with multiple dysplastic nevi that had these histologic features. Case reports Case 1. A 26-year-old white man had a pigmented lesion on his left arm that had been present for a few years and had become erythematous during the preceding few months. Physical examination revealed that the patient had multiple pigmented lesions distributed diffusely on his trunk and upper extremities. Many were greater than 5 mm in diameter and had irregular margins and variegated pigment. The lesion on the left arm was 6 mm in diameter, irregular in shape and pigment distribution, and surrounded by a rim of erythema. The clinical impression was that of an inflamed dysplastic nevus. This lesion and two other similarly dysplastic-appearing lesions were excised. Case 2. A 24-year-old white woman had multiple pigmented lesions on her face, trunk, and extremities. Her family history did not reveal melanoma or dysplastic nevus syndrome. The lesions were up to 8 mm in diameter. Many were variegated and had irregular margins and splaying of pigment. These lesions, thought to be dysplastic nevi, were excised. Histologic examination. Three lesions from each patient were examined. Each was greater than 0.6 mm in breadth, and the histologic features were similar in all biopsy specimens. There was lentiginous elongation of the rete ridges, with bridging of adjacent rete ridges by nests of atypical melanocyte~. Single melanocytes were also present along the dermoepidermal
From the Department of Pathology, Division of Dermatopathology,' and the Department of Medicine, Division of Dermatology,b New York Hospital-Cornell University Medical Center. Reprint requests: B. R. SmoJler, MD, Department of Pathology, Stanford University Medical Center, Stanford, CA 94305.
16/4/22902
Volume 24 Number 2, Part I February 1991
Brief communications 291
Fig. 1. Diffuse balloon cell transformation of large nests of melanocytes throughout three lesions examined. (X250.)
Fig. 2. Electron photomicrograph of affected melanocyte cytoplasm shows extensive vacuolization. Electron-dense material suggestive of melanin is present within some vacuoles (arrow). No well-formed melanosomes are identified. (X5800.) junction, but pagetoid upward migration ofmelanocytes was not evident. Within the dermis, nests ofsimilar a typical melanocytes had apparently maturcd and followed a course of progressive descent into the papillary dermis. At the deepest portions of the lesions, the nevus nests were slightly smaller, and the individ ual cells had somewhat less cytoplasm than did those in the epidermis and papillary dermis. However, they retained a great amount of cytoplasm that appeared to be vacuolated. The melanocytes had moderate cytologic atypia, with enlarged pleomorphic nuclei, expanded chromatin, and focally prominent nucleoli. The majority of melanocytes, in all of the lesions we stUdied, showed ballooning transformation characterized by
abundant vacuolated, pale-staining cytoplasm, with focal granular, cytoplasmic melanin pigment (Fig. I). Individual melanocytes were greater than 20 .urn in diameter. Mitoses were not observed in the dermal component of the lesions. Examination by electron microscopy of one of the lesions showed mclanocytes with diffuse cytoplasmic vacuolization. Some of the vacuoles contained electron-dense material suggestive of melanin (Fig. 2). There were no recognizable melanosomes in any nevus cells.
Discussion. Small foci of balloon cell transformation are seen in up to 2% of melanocytic nevi. 4 However. bal~
Journal of the American Academy of Dermatology
292 Briefcommunications loon cell transformation in more than 50% of the cells of a nevus is a relatively rare occurrence. 5 Goette and Doty6 found a predominance of balloon cells in 31 lesions classified as either compound or intradermal nevi, and additional rare cases within malignant melanomas have been reported. 2 The majority of reported cases have been in patients less than 30 years of age. 6 To our knowledge, extensive balloon cell transformation within a dysplastic nevus has not been described. In the present cases, we have shown the classically described changes that involved virtually all me1anocytes within multiple dysplastic nevi. Hashimoto and Bale3 have suggested that the ballooning transformation is caused by the progressive enlargement, degeneration, and coalescence ofmelanosomes and that melanization is thus largely prevented within affected melanocytes. Okun et at 7 found similar ultrastructural abnormalities, along with vacuolated melanosomes that had been transferred to keratinocytes. These findings would suggest that our patients have a melanocyte disorder in which there is altered melanization, in addition to the standard changes seen within dysplastic nevi. Given the architectural and cytologic disorder that occurs to varying degrees within dysplastic nevi, it is surprising that functional disorders of melanin biosynthesis do not occur more frequently in the more severely dysplastic melanocytic nevi. REFERENCES 1. Lever WF, Schaumburg-Lever G. Histopathology of the skin. 6th ed. Philadelphia: JB Lippincott, 1983:688. 2. Gardner W Alr, Vasquez MD. Balloon cell melanoma. Arch Pathol 1970;89:470-2. 3. Hashimoto K, Bale GF. An electron microscopic study of balloon ceJl nevus. Cancer 1972;30:530-40. 4. Gartmann H. Uber blasige Zellen in Naevuszellnaevus. Z Haut Geschlectskr 1960;28:148-59. 5. Schrader WA, Helwig EB. Balloon cell nevi. Cancer 1967;20: 1502-14.
6. Goette DK, Doty RD. Balloon cell nevus. Arch Dermatol 1978;114:109-11.
7. Okun MR, Donnellan B, Edelstein L. An ultrastructural study of balloon cell nevus. Cancer 1974;34:615-25.
A novel cryosurgical technique for gingival depigmentation Haim Tal, DMD, PhD* Tel Aviv, Israel Melanin pigmentation, carotene, and reduced hemoglobin and oxyhemoglobin all contribute to the normal From the Department of Periodontology, The Maurice and Gabriela Goldschlcger School of Dental Medicine, Tel Aviv University. Reprint requests: Dr. Haim TaJ; Department of Periodontology, Tel Aviv University, Ramat-Aviv 69978, Tel Aviv, Israel. 'Chairman. 16/4/22903
color of the oral mucosa.!' 2 However, melanin pigmentation of the oral mucosa has both diagnostic and anthropologic significance. 3, 4 The gingivae are the most frequently pigmented intraoral tissues, 2 and, contrary to general opinion, gingival pigmentation is not confined to the black population. Members of other nationalities (French, Filipino, Arabian, Chinese, Indian, German, Italian, Jewish, Greek, and Romanian) have gingival pigmentation. 5 Although physiologic gingival melanin pigmentation is not a medical problem, complaints of "black gums" are common, and requests for cosmetic therapy often come from fair-skinned persons with moderate or severe gingival pigmentation. The literature about depigmentation and repigmentation after surgical removal of pigmented gingivae in human beings is limited. 6, 7 The present study is based on a 2- to 5-year clinical observation after superficial cryosurgical treatment of moderate-to-heavily pigmented gingivae in seven patients. Material and methods. Seven nonsmoking patients (four women, three men) with moderate-to-heavy gingival melanin pigmentation were included in this study. All reported dark gums and requested "cosmetic therapy" to convert their "black gums" to "normal." Three patients were fair-skinned Ashkenazi Jews, and four were of Sephardic origin. Tissue reaction to cryosurgery was initially tested in a limited area of the gingiva in each patient. The treated sites were exposed to a gas-expansion cryoprobe (Nasal cryoprobe No. 2226, Frigitronics, Shelton, Conn.) cooled to -81 0 C for 10 seconds. The frozen site thawed spontaneously within 1 minute, and mild erythema soon developed.
Some pain was associated with the procedure, but most patients preferred to continue treatment without anesthesia; however, topical lidocaine ointment and local anesthesia were used for two patients. Treatment was limited to visible areas (medial to the first molar teeth). Clinical photographs were taken perioOically. Results. Superficial necrosis became apparent during the first week after freezing. A whitish slough separated from the underlying tissue, leaving a clean, granulated surface. At a 2 weeks, the treated site was covered with epithelium and between 3 and 4 weeks, keratinization was complete, and gingival stippling reformed. The treated gingiva was similar in color to nonpigmented gingiva (Fig. 1). Follow-up observations, during the first 3 years after treatment, did not show significant change in five of the patients. In two patients limited areas (3 X 5 mm and 4 X 4 mm) were slightly darker than the surrounding gingiva, but without cosmetic significance (Fig. 1, B). Patients did not report side effects, nor did they require additional treatment during the 5-year period after surgery. Discussion. Cryosurgery is an effective method for tissue cell devitalization. 8•13 The lethal mechanisms of freezing include the formation ofintracellular ice crystals, cell dehydration, and shrinkage. Cryosurgery denatures
Journal of the American Academy of Dermatology
Brief communications
ative, associated scalp lesions may be seen in 30% of lupus erythematosus profundus cases.6 Periorbital and eyelid edema resulting from lupus erythematosus profundus has been reported in the ophthalmologic literature. In one report,? persistent unilateral eyelid edema was the first recognized sign of lupus erythematosus profundus in three patients, and one case was associated with proptosis. These patients' symptoms improved in response to antimalarial therapy, and all had lesions of discoid LE, which were not present in our patient. One other report described unilateral proptosis in a case of systemic LE with associated swelling of the periorbital tissues. s An explanation for the proptosis was not given, although the clinical description was consistent with that of lupus erythematosus profundus. Another case of unilateral periorbital edema associated with ipsilateral lupus erythematosus profundus of the cheek has been reported in the dental literature. In this case, the trauma of oral surgery possibly exacerbated the panniculitis, and it was postulated that lymphatic obstruction, rather than actual involvement of the periorbital fat, had caused the periorbital edema. Although trauma apparently was not a contributing factor in our case, it has preceded other cases oflupus erythematosus profundus. I. 2, 10 In addition to the ocular findings in our patient, two other aspects of the case are worthy of comment. Initial lesions of lupus erythematosus profundus are usually freely movable. I , \0 Our patient's facial nodule on the left side was so indurated and fixed that we were concerned about the possibility of a neoplasm, although we were aware ofthe patient's past history oflupus erythematosus profundus. In addition, our patient had a long-standing nonscarring patchy hair loss that was clinically consistent with alopecia areata, which progressed despite treatment with antimalarial agents. This emphasizes that normalappearing skin with no clinically detectable depressions or indurations may overlie lesions of lupus erythematosus profundus. REFERENCES 1. Izumi AK, Takiguchi P. Lupus erythematosus panniculitis. Arch Dermatol 1983;119:61-4. 2. Tuffanelli DL. Lupus erythematosus (panniculitis) profundus: a classic revisited--eommentary and report of22 cases. Hawaii Med J 1982;41:394-7. 3. Zweiman B, Tomar RH, Gross PRo Lupus erythematosus profundus following thrombocytopenic purpura. Arch Dermatol 1975;111 :347-51. 4. Winkelmann RK. Panniculitis in connective tissue disease. Arch Dermatol1983;ll9:336-44. 5. Diaz-Jouanen E, DeHoratius RJ, Alarcon-Segovia D, et al. Systemic lupus erythematosus presenting as panniculitis (lupus profundus). Ann Intern Med 1975;82:376-9. 6. Sanchez NP, Peters MS, Winkelmann RK. The histopathology of lupus erythematosus panniculitis. J AM ACAD DERMATOL 1981;5:673-80. 7. Nowinski T, Bernardino V, Naidoff M, et al. Ocular involvement in lupus erythematosus profundus (panniculitis). Ophthalmology 1982;89:1149-54.
8. Brenner EH, Shock JP. Proptosis secondary to systemic lupus erythematosus. Arch OphthalmoI1974;91:81-2. 9. Klein SA, Tomaro AJ. Lupus erythematosus profundus: review of the literature and report of case. 1 Oral Surg 1975;33:454-7. 10. Izumi AK. Lupus erythematosus panniculitis. Clin DermatoI1985;3:69-77.
Balloon cell transformation in multiple dysplastic nevi Bruce R. Smoller, MD,a.b Susan Kindel, MD,b N. Scott McNutt, MD,a.b Mark H. Gray,a and Amy Hsua New York. New York Melanocytic nevi composed entirely of balloon cells are rare, I as are balloon cell melanomas? Balloon cells are melanocytes with extensive, pale-staining, and vacuolated cytoplasm. This histologic pattern is thought to be caused by a defect in melanosome formation that leads to faulty melanin production. 3 Although focal ballooning transformation is not uncommon, extensive change has not been reported in a dysplastic nevus. We recently saw two patients with multiple dysplastic nevi that had these histologic features. Case reports Case 1. A 26-year-old white man had a pigmented lesion on his left arm that had been present for a few years and had become erythematous during the preceding few months. Physical examination revealed that the patient had multiple pigmented lesions distributed diffusely on his trunk and upper extremities. Many were greater than 5 mm in diameter and had irregular margins and variegated pigment. The lesion on the left arm was 6 mm in diameter, irregular in shape and pigment distribution, and surrounded by a rim of erythema. The clinical impression was that of an inflamed dysplastic nevus. This lesion and two other similarly dysplastic-appearing lesions were excised. Case 2. A 24-year-old white woman had multiple pigmented lesions on her face, trunk, and extremities. Her family history did not reveal melanoma or dysplastic nevus syndrome. The lesions were up to 8 mm in diameter. Many were variegated and had irregular margins and splaying of pigment. These lesions, thought to be dysplastic nevi, were excised. Histologic examination. Three lesions from each patient were examined. Each was greater than 0.6 mm in breadth, and the histologic features were similar in all biopsy specimens. There was lentiginous elongation of the rete ridges, with bridging of adjacent rete ridges by nests of atypical melanocyte~. Single melanocytes were also present along the dermoepidermal
From the Department of Pathology, Division of Dermatopathology,' and the Department of Medicine, Division of Dermatology,b New York Hospital-Cornell University Medical Center. Reprint requests: B. R. SmoJler, MD, Department of Pathology, Stanford University Medical Center, Stanford, CA 94305.
16/4/22902
Volume 24 Number 2, Part I February 1991
Brief communications 291
Fig. 1. Diffuse balloon cell transformation of large nests of melanocytes throughout three lesions examined. (X250.)
Fig. 2. Electron photomicrograph of affected melanocyte cytoplasm shows extensive vacuolization. Electron-dense material suggestive of melanin is present within some vacuoles (arrow). No well-formed melanosomes are identified. (X5800.) junction, but pagetoid upward migration ofmelanocytes was not evident. Within the dermis, nests ofsimilar a typical melanocytes had apparently maturcd and followed a course of progressive descent into the papillary dermis. At the deepest portions of the lesions, the nevus nests were slightly smaller, and the individ ual cells had somewhat less cytoplasm than did those in the epidermis and papillary dermis. However, they retained a great amount of cytoplasm that appeared to be vacuolated. The melanocytes had moderate cytologic atypia, with enlarged pleomorphic nuclei, expanded chromatin, and focally prominent nucleoli. The majority of melanocytes, in all of the lesions we stUdied, showed ballooning transformation characterized by
abundant vacuolated, pale-staining cytoplasm, with focal granular, cytoplasmic melanin pigment (Fig. I). Individual melanocytes were greater than 20 .urn in diameter. Mitoses were not observed in the dermal component of the lesions. Examination by electron microscopy of one of the lesions showed mclanocytes with diffuse cytoplasmic vacuolization. Some of the vacuoles contained electron-dense material suggestive of melanin (Fig. 2). There were no recognizable melanosomes in any nevus cells.
Discussion. Small foci of balloon cell transformation are seen in up to 2% of melanocytic nevi. 4 However. bal~
Journal of the American Academy of Dermatology
292 Briefcommunications loon cell transformation in more than 50% of the cells of a nevus is a relatively rare occurrence. 5 Goette and Doty6 found a predominance of balloon cells in 31 lesions classified as either compound or intradermal nevi, and additional rare cases within malignant melanomas have been reported. 2 The majority of reported cases have been in patients less than 30 years of age. 6 To our knowledge, extensive balloon cell transformation within a dysplastic nevus has not been described. In the present cases, we have shown the classically described changes that involved virtually all me1anocytes within multiple dysplastic nevi. Hashimoto and Bale3 have suggested that the ballooning transformation is caused by the progressive enlargement, degeneration, and coalescence ofmelanosomes and that melanization is thus largely prevented within affected melanocytes. Okun et at 7 found similar ultrastructural abnormalities, along with vacuolated melanosomes that had been transferred to keratinocytes. These findings would suggest that our patients have a melanocyte disorder in which there is altered melanization, in addition to the standard changes seen within dysplastic nevi. Given the architectural and cytologic disorder that occurs to varying degrees within dysplastic nevi, it is surprising that functional disorders of melanin biosynthesis do not occur more frequently in the more severely dysplastic melanocytic nevi. REFERENCES 1. Lever WF, Schaumburg-Lever G. Histopathology of the skin. 6th ed. Philadelphia: JB Lippincott, 1983:688. 2. Gardner W Alr, Vasquez MD. Balloon cell melanoma. Arch Pathol 1970;89:470-2. 3. Hashimoto K, Bale GF. An electron microscopic study of balloon ceJl nevus. Cancer 1972;30:530-40. 4. Gartmann H. Uber blasige Zellen in Naevuszellnaevus. Z Haut Geschlectskr 1960;28:148-59. 5. Schrader WA, Helwig EB. Balloon cell nevi. Cancer 1967;20: 1502-14.
6. Goette DK, Doty RD. Balloon cell nevus. Arch Dermatol 1978;114:109-11.
7. Okun MR, Donnellan B, Edelstein L. An ultrastructural study of balloon cell nevus. Cancer 1974;34:615-25.
A novel cryosurgical technique for gingival depigmentation Haim Tal, DMD, PhD* Tel Aviv, Israel Melanin pigmentation, carotene, and reduced hemoglobin and oxyhemoglobin all contribute to the normal From the Department of Periodontology, The Maurice and Gabriela Goldschlcger School of Dental Medicine, Tel Aviv University. Reprint requests: Dr. Haim TaJ; Department of Periodontology, Tel Aviv University, Ramat-Aviv 69978, Tel Aviv, Israel. 'Chairman. 16/4/22903
color of the oral mucosa.!' 2 However, melanin pigmentation of the oral mucosa has both diagnostic and anthropologic significance. 3, 4 The gingivae are the most frequently pigmented intraoral tissues, 2 and, contrary to general opinion, gingival pigmentation is not confined to the black population. Members of other nationalities (French, Filipino, Arabian, Chinese, Indian, German, Italian, Jewish, Greek, and Romanian) have gingival pigmentation. 5 Although physiologic gingival melanin pigmentation is not a medical problem, complaints of "black gums" are common, and requests for cosmetic therapy often come from fair-skinned persons with moderate or severe gingival pigmentation. The literature about depigmentation and repigmentation after surgical removal of pigmented gingivae in human beings is limited. 6, 7 The present study is based on a 2- to 5-year clinical observation after superficial cryosurgical treatment of moderate-to-heavily pigmented gingivae in seven patients. Material and methods. Seven nonsmoking patients (four women, three men) with moderate-to-heavy gingival melanin pigmentation were included in this study. All reported dark gums and requested "cosmetic therapy" to convert their "black gums" to "normal." Three patients were fair-skinned Ashkenazi Jews, and four were of Sephardic origin. Tissue reaction to cryosurgery was initially tested in a limited area of the gingiva in each patient. The treated sites were exposed to a gas-expansion cryoprobe (Nasal cryoprobe No. 2226, Frigitronics, Shelton, Conn.) cooled to -81 0 C for 10 seconds. The frozen site thawed spontaneously within 1 minute, and mild erythema soon developed.
Some pain was associated with the procedure, but most patients preferred to continue treatment without anesthesia; however, topical lidocaine ointment and local anesthesia were used for two patients. Treatment was limited to visible areas (medial to the first molar teeth). Clinical photographs were taken perioOically. Results. Superficial necrosis became apparent during the first week after freezing. A whitish slough separated from the underlying tissue, leaving a clean, granulated surface. At a 2 weeks, the treated site was covered with epithelium and between 3 and 4 weeks, keratinization was complete, and gingival stippling reformed. The treated gingiva was similar in color to nonpigmented gingiva (Fig. 1). Follow-up observations, during the first 3 years after treatment, did not show significant change in five of the patients. In two patients limited areas (3 X 5 mm and 4 X 4 mm) were slightly darker than the surrounding gingiva, but without cosmetic significance (Fig. 1, B). Patients did not report side effects, nor did they require additional treatment during the 5-year period after surgery. Discussion. Cryosurgery is an effective method for tissue cell devitalization. 8•13 The lethal mechanisms of freezing include the formation ofintracellular ice crystals, cell dehydration, and shrinkage. Cryosurgery denatures