Generalized Smooth Muscle Hamartoma with Multiple Congenital Anomalies without the “Michelin Tire Baby” Phenotype

Janicke, Elise C.; Nazareth, Michael R.; Rothman, Ilene L.

doi:10.1111/pde.12280

Cited by 9 publications

(3 citation statements)

References 11 publications

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“…Although the etiopathogenesis of the SMH remains unclear, cases can be divided into two groups: congenital 1,5,9,10,16‐24 and acquired SMH 2‐4,6‐8,11,12,15,25‐32 . Eleven of our cases (41%) presented as a congenital lesion, most commonly located on the torso and lower extremity (six cases, 55%) followed by the upper extremity (two cases, 18%) and the head and neck area (two cases, 18%), and one case (9%) was located on the penile shaft.…”

Section: Discussionmentioning

confidence: 88%

Smooth muscle hamartoma and striated muscle hamartoma: Clinicopathologic characterization of two rare entities and literature review

González

Dehner

2020

J Cutan Pathol

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Smooth muscle hamartoma (SMH) and striated muscle hamartoma (STH) are anomalous proliferations of smooth muscle or striated muscle, respectively, in anatomic sites where these tissues are normally present. To date, only limited cases have been reported describing these lesions. In this study, we sought to characterize the clinicopathologic features of both SMH and STH. A total of 27 cases of SMH and 12 cases of STH from 1990 to 2020 were identified. SMH cases had a slight male predominance (63%) and a mean age of presentation of 20 years (range: 4 months‐91 years), with a mean size of 9.3 mm (±13.3). In contrast, STH were equally distributed in gender, with a mean age of presentation of 40 years (range: 3 months‐66 years) and a mean size of 5.7 mm (±3.6). SMH were more commonly located in the torso and extremities (70%) and STH in the head and neck area (92%). One case of SMH recurred after 1.1 years and in the initial diagnosis the lesion was present at the tissue edge. None of the cases of STH had a recurrence. We present the largest cohort of SMH and STH, and report the first case of a recurrent SMH, suggesting the importance of obtaining a clean margin for these lesions.

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Section: Discussionmentioning

confidence: 88%

Smooth muscle hamartoma and striated muscle hamartoma: Clinicopathologic characterization of two rare entities and literature review

González

Dehner

2020

J Cutan Pathol

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“…Generalized SMHs have been reported to raise the question of an overgrowth syndrome. 367 Bundles of disorganized smooth muscle originating from the arrector pili muscle occupy the dermis with the formation of a dermal occupying mass ( Figure 43). The blue nevus and SMH are rarely seen together in the same lesion.…”

Section: Sarcomas and Smooth Muscle Tumorsmentioning

confidence: 99%

Nonepithelial Tumors and Tumor-like Lesions of the Skin and Subcutis in Children

Dehner

Gru

2018

Pediatr Dev Pathol

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This overview of mesenchymal tumors presenting in the skin and/or subcutis in children brings together the range of neoplasms and hamartomas which are seen in this age-group. It is not surprising from the perspective of the pediatric or general surgical pathologist that vascular anomalies, including true neoplasms and vascular malformations, are the common phenotypic category. Since there is considerable morphologic overlap among these lesions, clinicopathologic correlation may be more important than for many of the other mesenchymal tumors. The skin and subcutis are the most common sites of clinical presentation for the infantile myofibroma which is the most common of fibrous mesenchymal tumors in children. Several of the other mesenchymal tumors are more common adults-like dermatofibrosarcoma protuberans, but nonetheless have an important presence in children, even as a congenital neoplasm. A lipomatous tumor in a young child should be considered as a possible manifestation of an overgrowth syndrome.

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“…At the bottom of Table are listed the number and combination of criteria necessary for a diagnosis of CSFS, as well as those necessary for a probable diagnosis. The case of a baby with generalized smooth muscle hamartoma with multiple congenital anomalies without the MTB phenotype was recently reported and it was noted that she could possibly fit in this group of patients with the MTB phenotype. It was proposed that the MTB phenotype might not be a necessary component of this constellation of findings.…”

Section: Proposed Criteria For a Syndromementioning

confidence: 99%

Michelin Tire Baby Syndrome: A Review of the Literature and a Proposal for Diagnostic Criteria with Adoption of the Name Circumferential Skin Folds Syndrome

Rothman

2014

Pediatric Dermatology

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The term Michelin tire baby (MTB), named for the cartoon mascot of the Michelin Tire Company, has been used to describe babies with multiple symmetric circumferential rings of folded skin. In those reported with this phenotype who had skin biopsies, pathology has shown nevus lipomatosis, smooth muscle hamartoma, degenerative collagen, and scarring. Others did not undergo biopsy or had normal skin. Many individuals with the MTB phenotype have had a variety of other congenital anomalies. I review the literature on MTB and the history of the designation Michelin tire baby Syndrome (MTBS). Because the term MTBS has been poorly defined or not defined at all, I propose strict criteria for diagnosis. In doing so, it is recommended that the syndrome be renamed to avoid further confusion.

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Generalized Smooth Muscle Hamartoma with Multiple Congenital Anomalies without the “Michelin Tire Baby” Phenotype

Cited by 9 publications

References 11 publications

Smooth muscle hamartoma and striated muscle hamartoma: Clinicopathologic characterization of two rare entities and literature review

Smooth muscle hamartoma and striated muscle hamartoma: Clinicopathologic characterization of two rare entities and literature review

Nonepithelial Tumors and Tumor-like Lesions of the Skin and Subcutis in Children

Michelin Tire Baby Syndrome: A Review of the Literature and a Proposal for Diagnostic Criteria with Adoption of the Name Circumferential Skin Folds Syndrome

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