2007
DOI: 10.1111/j.1525-1470.2007.00551.x
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Myokymia as a Presenting Sign of Congenital Smooth Muscle Hamartoma

Abstract: 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 derm… Show more

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Cited by 26 publications
(12 citation statements)
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“…The first consists of transient elevation of the lesion with piloerection and sometimes whitening after rubbing, and myokymia refers to spontaneous transient episodes of vermiform movement of the involved area. 4,5 Our case represents an unusual form of SMH. The lesion closely mimicked cutis marmorata telangiectatica congenita (CMTC) and even contained atrophic areas.…”
Section: Discussionmentioning
confidence: 76%
See 1 more Smart Citation
“…The first consists of transient elevation of the lesion with piloerection and sometimes whitening after rubbing, and myokymia refers to spontaneous transient episodes of vermiform movement of the involved area. 4,5 Our case represents an unusual form of SMH. The lesion closely mimicked cutis marmorata telangiectatica congenita (CMTC) and even contained atrophic areas.…”
Section: Discussionmentioning
confidence: 76%
“…Further clues for SMH are the pseudo‐Darier sign and myokymia. The first consists of transient elevation of the lesion with piloerection and sometimes whitening after rubbing, and myokymia refers to spontaneous transient episodes of vermiform movement of the involved area …”
Section: Discussionmentioning
confidence: 99%
“…Prior to this seminal report, very few cases of a similar phenomenon had been described in the literature . Early reports postulated hyperplastic smooth muscle as the underlying etiology for the observed clinical syndrome, occasionally leading to provisional diagnoses of congenital smooth muscle hamartoma (CSMH) . In such cases, histologic examination purportedly revealed hyperplastic smooth muscle tissue, though further review of the pathologic images called into question the true degree of hyperplasia .…”
Section: Discussionmentioning
confidence: 98%
“…The term infantile transient smooth muscle contraction of the skin (ITSMC) was initially coined in 2013 by Torrelo et al when describing a series of nine infants who experienced “transient rippling of the skin” in response to various stimuli . Prior to this seminal report, very few cases of a similar phenomenon had been described in the literature . Early reports postulated hyperplastic smooth muscle as the underlying etiology for the observed clinical syndrome, occasionally leading to provisional diagnoses of congenital smooth muscle hamartoma (CSMH) .…”
Section: Discussionmentioning
confidence: 99%
See 1 more Smart Citation