Congenital Smooth Muscle Hamartoma of the Skin

Metzker, Aryeh; Amir, Jacob; Rotem, Arie; Merlob, Paul

doi:10.1111/j.1525-1470.1984.tb00441.x

Cited by 31 publications

(19 citation statements)

References 8 publications

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“…Conversely, unlike BN, hypertrichosis and hyperpigmentation often decrease with age in CSMH. 15,16 The key features of CSMH include smooth muscle hyperplasia of the reticular dermis with variably oriented, well-defined bundles of smooth muscle. 1,5,6,17,18 BN also frequently exhibits dermal smooth muscle proliferation on histological examination but does not appear as a mass or bundle-like structure.…”

Section: Discussionmentioning

confidence: 99%

Clinicopathologic characteristics of early‐onset Becker's nevus in Korean children and adolescents

et al. 2017

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Section: Discussionmentioning

confidence: 99%

Clinicopathologic characteristics of early‐onset Becker's nevus in Korean children and adolescents

et al. 2017

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“…Some authors suggest that the condition may represent an organoid connective tissue hamartoma with at least three components: smooth muscle bundles, nerve fibers, and prominent vellus hair (6). The hypothesis of an organoid nevus is also supported by the coordinated rippling movements occasionally observed (9,11,20) which suggest that the lesion may act as a functional unit.…”

Section: Discussionmentioning

confidence: 99%

Myokymia as a Presenting Sign of Congenital Smooth Muscle Hamartoma

Kienast

Maerker

Hoeger

2007

Pediatric Dermatology

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We report a 4-week-old male infant with intermittent asymptomatic bouts of vermicular movements of the skin (myokymia). Myokymia started at the age of 4 days. Attacks occurred approximately every 30-60 minutes and could be elicited by gently stroking the skin. Macroscopically no abnormalities typical for smooth muscle hamartoma such as hypertrichosis or hyperpigmentation were observed. Histologic examination of a skin biopsy specimen showed bundles of hyperplastic smooth muscle tissue within the reticular dermis which were associated with hair follicles. Karyotyping did not reveal any structural or numeric chromosomal anomaly. The findings led to the diagnosis of congenital smooth muscle hamartoma. On follow-up at the age of 24 months myokymia persisted, but with a marked reduction in the number of attacks per day. The phenomenon of myokymia results from neurally induced spontaneous contraction of dermal smooth muscles. It is also known as "pseudo-Darier's sign." While most of those affected previously reported had hypertrichosis or hyperpigmentation, this patient seems to be the first to present with myokymia alone.

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“…Although most frequently encountered as a distinct clinicopathologic entity, histoiogic findings in CSMH have been described in Becker nevus (1) and the Michelin tire syndrome (2^). Recent reports (5)(6)(7)(8)(9)(10)(11)(12)(13)(14) suggest that CSMH may be more common than once thought, yet frequently is not recognized. It occurs more frequently in males then females at a ratio of approximately 2:1 (5)(6)(7)(8)(9)(10)(11)(12)(13)(14).…”

Section: Discussionmentioning

confidence: 99%