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Acquired cutaneous smooth muscle hamartoma
Acquired cutaneous smooth muscle hamartoma Thomas N. Darling, MD, PhD,a Hideko Kamino, MD,b and John C. Murray, MDa
Durham, North Carolina A 35-year-old white man had an indurated, indistinct plaque on the anterior aspect of the
neck for 10 years. Results of biopsyspecimens showed an excessof smooth muscle bundles scattered throughout the dermis. Unlike previously reported cases of acquired smooth muscle hamartomas, it did not occur in association with a Becker nevus. (J AM ACAD DERMATaL 1993;28:844-5.)
Cutaneous smooth muscle hamartomas are benign proliferations ofsmooth muscle bundles within the dermis. They can be congenital or acquired. Congenital smooth muscle hamartomas are fleshcolored or slightly hyperpigmented plaques that often contain long vellus hairs. 1, 2 Acquired smooth muscle hamartomas have been reported most frequently in association with a Becker nevus. 3- 5 A Becker nevus is an organoid hamartoma characterized by an increase in pigment in the epidermal basal layer and the presence of terminal hairs." We describe an acquired smooth muscle hamartoma that does not show hyperpigmentation or increased hairs.
CASE REPORT A 35-year-old white man had an erythematous plaque on his neck for about 10years. It gradually became more prominent. The plaque was asymptomatic except for pruritus after a hot shower. Examination revealed an erythematous, indistinct, indurated plaque that extended in a band across the lower part of the neck (Fig. 1). There was no hyperpigmentation or hypertrichosis. A pseudo-Darier's sign could not be elicited. Findings of a skin biopsy specimen revealed increased smooth muscle bundles scattered throughout the dermis (Fig. 2, A). These smooth muscle bundles were in varying orientations without an appreciable relation to the hairs. The presence of increased smooth muscle fibers was confirmed with Masson's trichrome stain (Fig. 2, B).
DISCUSSION To the best of our knowledge, this is the first report of an acquired smooth muscle hamartoma From the Division of Dermatology, Department of Medicine" and Department of Pathology," Duke University Medical Center. Reprints not available. Copyright (D 1993 by the American Academy of Dermatology. Inc. 0190-9622/93 $1.00+ .10
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without increased pigment or hairs. The most similar case is an acquired smooth muscle hamartoma that was hyperpigmented,? Most other acquired smooth muscle hamartomas are observed in association with a Becker nevus. 3-5Proliferations of smooth muscle have also been reported in association with seborrheic keratoses.' nevus sebaceus, and blue nevi.! Several features distinguish this lesion from a leiomyoma. The scattered pattern of smooth muscle bundles throughout the dermis is typical of smooth muscle hamartomas. The smooth muscle bundles in leiomyomas are more tightly interlacing. Grossly, leiomyomas appear as discrete nodules, sometimes with a red-brown color. They are also painful or tender.v'? Congenital smooth muscle hamartomas are often slightly hyperpigmented and contain increased velIus hairs. I, 2, II The degree of pigmentation and number of hairs are variable. Many are fleshcolored, t, 12 and some may show only an increase in smooth muscle bundles.P: 14 In contrast, a Becker nevus develops later in life and is characterized by hyperpigmentation and usually by hypertrichosis. Hyperplasia of smooth muscle in the dermis has been reported by some authors-' but not by others. 6• 15 Because of the overlap of features, some have postulated that there is a spectrum of changes that can be seen between smooth muscle hamartomas and Becker nevi.'! 16-18 More recent reports have emphasized the unique features of congenitalsmooth muscle hamartomas compared with Becker nevi. 1, 2 It is probably appropriate to describe these lesions as distinct entities, each with a spectrum of hamartomatous growth that involves the epidermis (hyperpigmentation), hair follicles (hypertrichosis), and dermis (smooth muscle). This avoids the problem of having to group the present case with Becker nevi.
Journal of the American Academy of Dermatology Volume 28, Number 5, Part 2
Darling et al. 845
Fig. 1. Indistinct plaque extending in a band across the mid neck and down to the supraclavicular region. Fig. 2. Proliferation of bundles of smooth muscle within the dermis. (A, Hematoxylin-eosin stain; B, Masson's trichrome stain.)
Funding for publication ofthe color photographs in this article was provided by Schering Laboratories. REFERENCES 1. Hanke CW, O'Brian JJ, Peters WC, et a!' Congenital smooth muscle hamartoma masquerading as congenital pigmented nevus. J Derrnatol Surg Oncol 1985;11: 714-7. 2. Zvulunov A, Rotem A, MerlobP, et al. Congenital smooth muscle hamartoma. Am J Dis Child 1990;144:782-4. 3. HanekeE. Thedermal component inmelanosis naeviformis Becker. J Cutan Pathol 1979;6:53-8. 4. Stokes JH. Nevus pilaris with hyperplasia of nonstriated muscle. Arch Dermatol 1923;7:479-81. 5. Urbanek RW, Johnson We. Smooth muscle hamartoma associated with Becker's nevus. Arch Dermatol 1978; 114:104-6. 6. Glinick SE, Alper JC, Bogaars H, et al. Becker's melanosis: associated abnormalities. J AM ACAD DERMATOL 1983;9:509-14. 7. Wong RC, Solomon AR. Acquired dermalsmooth-muscle hamartoma. Cutis 1985;35:369-70. 8. Montgomery H, Winkelman RK. Smooth-muscle tumors of the skin. Arch Dermatol 1959;79:32-4l.
9. Fisher we, Helwig EB. Leiomyomas of the skin. Arch Dermatol 1963;88:78-88. 10. From L, Assaad D. Neoplasms and hyperplasia of neural and muscularorigin. In: FitzpatrickTB, EisenAZ, WolfK, et aI., eds. Dermatology in general medicine. New York: McGraw-Hill, 1987:1053-4. 11. Slifman NR, Harrist TJ, Rhodes AR. Congenitalarrector pili hamartoma. Arch Dermatol 1965;121:1034-7. 12. Bronson OM, Fretzin DF, Farrell LN. Congenitalpilarand smoothmusclenevus.JAMAcADDERMATOL 1983;8:111-4. 13. Huffman DW, MallorySB. Congenitalsmooth musclehamartoma. Am Fam Physician 1989;39:117-20. 14. Plewig G, Schmoeckel C. Naevus musculi arrector pili. Hautarzt 1979;30:503-5. 15. Becker SW. Concurrent melanosis and hypertrichosis in distribution of nevus unius lateris. Arch Derrnatol Syph 1949;60:155-60.
16. Karo KR, Gange RW. Smooth-musclehamartoma: possib1e congenital Becker's nevus. Arch Dermatol 1981; 117:678-9. 17. Chapel TA, Tavafoghi Y, Mehregan AH"et al. Becker's melanosis: an organoidhamartoma. Cutis 1981;27:405-15. 18. Civatte J, Marinho E, OliverSantos R. Hamartoma muscular lisa 0 nevo de Becker? A proposito de cuatro casas. Med Cutan Ibero Lat Am 1988;16:145-8.
Durham, North Carolina A 35-year-old white man had an indurated, indistinct plaque on the anterior aspect of the
neck for 10 years. Results of biopsyspecimens showed an excessof smooth muscle bundles scattered throughout the dermis. Unlike previously reported cases of acquired smooth muscle hamartomas, it did not occur in association with a Becker nevus. (J AM ACAD DERMATaL 1993;28:844-5.)
Cutaneous smooth muscle hamartomas are benign proliferations ofsmooth muscle bundles within the dermis. They can be congenital or acquired. Congenital smooth muscle hamartomas are fleshcolored or slightly hyperpigmented plaques that often contain long vellus hairs. 1, 2 Acquired smooth muscle hamartomas have been reported most frequently in association with a Becker nevus. 3- 5 A Becker nevus is an organoid hamartoma characterized by an increase in pigment in the epidermal basal layer and the presence of terminal hairs." We describe an acquired smooth muscle hamartoma that does not show hyperpigmentation or increased hairs.
CASE REPORT A 35-year-old white man had an erythematous plaque on his neck for about 10years. It gradually became more prominent. The plaque was asymptomatic except for pruritus after a hot shower. Examination revealed an erythematous, indistinct, indurated plaque that extended in a band across the lower part of the neck (Fig. 1). There was no hyperpigmentation or hypertrichosis. A pseudo-Darier's sign could not be elicited. Findings of a skin biopsy specimen revealed increased smooth muscle bundles scattered throughout the dermis (Fig. 2, A). These smooth muscle bundles were in varying orientations without an appreciable relation to the hairs. The presence of increased smooth muscle fibers was confirmed with Masson's trichrome stain (Fig. 2, B).
DISCUSSION To the best of our knowledge, this is the first report of an acquired smooth muscle hamartoma From the Division of Dermatology, Department of Medicine" and Department of Pathology," Duke University Medical Center. Reprints not available. Copyright (D 1993 by the American Academy of Dermatology. Inc. 0190-9622/93 $1.00+ .10
844
16/4/44418
without increased pigment or hairs. The most similar case is an acquired smooth muscle hamartoma that was hyperpigmented,? Most other acquired smooth muscle hamartomas are observed in association with a Becker nevus. 3-5Proliferations of smooth muscle have also been reported in association with seborrheic keratoses.' nevus sebaceus, and blue nevi.! Several features distinguish this lesion from a leiomyoma. The scattered pattern of smooth muscle bundles throughout the dermis is typical of smooth muscle hamartomas. The smooth muscle bundles in leiomyomas are more tightly interlacing. Grossly, leiomyomas appear as discrete nodules, sometimes with a red-brown color. They are also painful or tender.v'? Congenital smooth muscle hamartomas are often slightly hyperpigmented and contain increased velIus hairs. I, 2, II The degree of pigmentation and number of hairs are variable. Many are fleshcolored, t, 12 and some may show only an increase in smooth muscle bundles.P: 14 In contrast, a Becker nevus develops later in life and is characterized by hyperpigmentation and usually by hypertrichosis. Hyperplasia of smooth muscle in the dermis has been reported by some authors-' but not by others. 6• 15 Because of the overlap of features, some have postulated that there is a spectrum of changes that can be seen between smooth muscle hamartomas and Becker nevi.'! 16-18 More recent reports have emphasized the unique features of congenitalsmooth muscle hamartomas compared with Becker nevi. 1, 2 It is probably appropriate to describe these lesions as distinct entities, each with a spectrum of hamartomatous growth that involves the epidermis (hyperpigmentation), hair follicles (hypertrichosis), and dermis (smooth muscle). This avoids the problem of having to group the present case with Becker nevi.
Journal of the American Academy of Dermatology Volume 28, Number 5, Part 2
Darling et al. 845
Fig. 1. Indistinct plaque extending in a band across the mid neck and down to the supraclavicular region. Fig. 2. Proliferation of bundles of smooth muscle within the dermis. (A, Hematoxylin-eosin stain; B, Masson's trichrome stain.)
Funding for publication ofthe color photographs in this article was provided by Schering Laboratories. REFERENCES 1. Hanke CW, O'Brian JJ, Peters WC, et a!' Congenital smooth muscle hamartoma masquerading as congenital pigmented nevus. J Derrnatol Surg Oncol 1985;11: 714-7. 2. Zvulunov A, Rotem A, MerlobP, et al. Congenital smooth muscle hamartoma. Am J Dis Child 1990;144:782-4. 3. HanekeE. Thedermal component inmelanosis naeviformis Becker. J Cutan Pathol 1979;6:53-8. 4. Stokes JH. Nevus pilaris with hyperplasia of nonstriated muscle. Arch Dermatol 1923;7:479-81. 5. Urbanek RW, Johnson We. Smooth muscle hamartoma associated with Becker's nevus. Arch Dermatol 1978; 114:104-6. 6. Glinick SE, Alper JC, Bogaars H, et al. Becker's melanosis: associated abnormalities. J AM ACAD DERMATOL 1983;9:509-14. 7. Wong RC, Solomon AR. Acquired dermalsmooth-muscle hamartoma. Cutis 1985;35:369-70. 8. Montgomery H, Winkelman RK. Smooth-muscle tumors of the skin. Arch Dermatol 1959;79:32-4l.
9. Fisher we, Helwig EB. Leiomyomas of the skin. Arch Dermatol 1963;88:78-88. 10. From L, Assaad D. Neoplasms and hyperplasia of neural and muscularorigin. In: FitzpatrickTB, EisenAZ, WolfK, et aI., eds. Dermatology in general medicine. New York: McGraw-Hill, 1987:1053-4. 11. Slifman NR, Harrist TJ, Rhodes AR. Congenitalarrector pili hamartoma. Arch Dermatol 1965;121:1034-7. 12. Bronson OM, Fretzin DF, Farrell LN. Congenitalpilarand smoothmusclenevus.JAMAcADDERMATOL 1983;8:111-4. 13. Huffman DW, MallorySB. Congenitalsmooth musclehamartoma. Am Fam Physician 1989;39:117-20. 14. Plewig G, Schmoeckel C. Naevus musculi arrector pili. Hautarzt 1979;30:503-5. 15. Becker SW. Concurrent melanosis and hypertrichosis in distribution of nevus unius lateris. Arch Derrnatol Syph 1949;60:155-60.
16. Karo KR, Gange RW. Smooth-musclehamartoma: possib1e congenital Becker's nevus. Arch Dermatol 1981; 117:678-9. 17. Chapel TA, Tavafoghi Y, Mehregan AH"et al. Becker's melanosis: an organoidhamartoma. Cutis 1981;27:405-15. 18. Civatte J, Marinho E, OliverSantos R. Hamartoma muscular lisa 0 nevo de Becker? A proposito de cuatro casas. Med Cutan Ibero Lat Am 1988;16:145-8.