Congenital smooth muscle hamartoma. A report of six cases and a review of the literature

Johnson, Molly D.; Jacobs, Alvin H.

doi:10.1001/archderm.125.6.820

Cited by 40 publications

(52 citation statements)

References 14 publications

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“…Histologically there is a band-like infiltrate of mature pilar smooth muscle extending from dermis to the superficial subcutis ( Figure 1). [1][2][3][4][5][6][7][8][9] Smooth muscle hamartoma of the breast parenchyma entails bundles of non-atypical smooth muscle present within the breast parenchyma between the lobules, often along with fibrous stroma and scattered fat cells. 10 …”

Section: Smooth Muscle Hamartomamentioning

confidence: 99%

Smooth muscle tumors of soft tissue and non-uterine viscera: biology and prognosis

Miettinen

2014

Modern Pathology

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Smooth muscle tumors are here considered an essentially dichotomous group composed of benign leiomyomas and malignant leiomyosarcomas. Soft tissue smooth muscle tumors with both atypia and mitotic activity are generally diagnosed leiomyosarcomas acknowledging potential for metastasis. However, lesions exist that cannot be comfortably placed in either category, and in such cases the designation 'smooth muscle tumor of uncertain biologic potential' is appropriate. The use of this category is often necessary with limited sampling, such as needle core biopsies. Benign smooth muscle tumors include smooth muscle hamartoma and angioleiomyoma. A specific category of leiomyomas are estrogen-receptor positive ones in women. These are similar to uterine leiomyomas and can occur anywhere in the abdomen and abdominal wall. Leiomyosarcomas can occur at any site, although are more frequent in the retroperitoneum and proximal extremities. They are recognized by likeness to smooth muscle cells but can undergo pleomorphic evolution ('dedifferentiation'). Presence of smooth muscle actin is nearly uniform and desmin-positivity usual. This and the lack of KIT expression separate leiomyosarcoma from GIST, an important problem in abdominal soft tissues. EBVassociated smooth muscle tumors are a specific subcategory occurring in AIDS or post-transplant patients. These tumors can have incomplete smooth muscle differentiation but show nuclear EBER as a diagnostic feature. In contrast to many other soft tissue tumors, genetics of smooth muscle tumors are poorly understood and such diagnostic testing is not yet generally applicable in this histogenetic group. Leiomyosarcomas are known to be genetically complex, often showing 'chaotic' karyotypes including aneuploidy or polyploidy, and no recurrent tumor-specific translocations have been detected.

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Section: Smooth Muscle Hamartomamentioning

confidence: 99%

Smooth muscle tumors of soft tissue and non-uterine viscera: biology and prognosis

Miettinen

2014

Modern Pathology

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“…We could not find a report of such a lesion in the literature. From a histogenetic standpoint, lesions most closely related to the one we report on are probably smooth muscle hamartomas associated with hair follicles 5,6 and the so-called congenital panfollicular nevus 7 . However, the former microscopically differ from our case in showing no abnormality of the hair follicle itself and in the absence of other adnexal differentiations, and the latter lacks a prominent mesenchymal component and predominantly shows follicular differentiation.…”

Section: Discussionmentioning

confidence: 65%

An unusual hamartoma of the folliculosebaceous‐apocrine unit: a case report

2006

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Hamartoma is a neoplasm-like lesion composed of an abnormal mixture of tissues native to the area. We report on an unusual example of a hamartomatous lesion that showed an abnormal mixture of elements of the folliculosebaceous-apocrine unit and arrector pili muscle. A 13-year-old female presented with two nodules situated 2 cm apart on the lower leg that were present since birth. The patient reported some enlargement and slight pain of the otherwise asymptomatic lesions during 2 years, before they were surgically removed. The patient obviously had no skin problems afterwards; her 12-year follow-up was significant for leucopenia and nephrolithiasis. The biopsies from both lesions looked identical. Each lesion was biphasic, i.e. composed of a predominant mesenchymal component and epithelial elements. The mesenchymal part resembled a leiomyoma, or in minor areas, the constituting cells had a myofibroblastic appearance. Encased within the mesenchymal component were large infundibulocystic structures and plentiful grouped and scattered roundish solid epithelial nodules, duct-like, glandular, and tubular elements. Some of the epithelial structures were reminiscent of those seen in tubular apocrine adenoma; others resembled syringomatous elements. We interpreted this unusual lesion as a hamartoma of the folliculosebaceous-apocrine unit.

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“…It is now widely accepted that Becker nevus and smooth muscle hamartoma are related conditions, reflecting ends of a spectrum of a hamartomatous process involving melanocytic, follicular, and smooth muscle elements. [23][24][25] Unlike its smooth muscle counterpart, an associated melanocytic abnormality, either as a component of the hamartomatous proliferation or otherwise, has never been described in RMH before.…”

Section: Discussionmentioning

confidence: 99%

Rhabdomyomatous Mesenchymal Hamartoma With Lentiginous Melanocytic Hyperplasia: An Inductive Phenomenon or Component of the Hamartoma?

Biswas

Pittius²

2008

The American Journal of Dermatopathology

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Rhabdomyomatous mesenchymal hamartoma (RMH) is a rare cutaneous hamartoma characterized by abundant bundles of mature skeletal muscle fibers admixed with other mesenchymal elements. We report a case of RMH in a 7-month-old girl with associated lentiginous melanocytic hyperplasia. The melanocytic hyperplasia was not apparent clinically and was only evident on histological examination. Unlike a smooth muscle hamartoma, melanocytic abnormalities have never been described in the context of RMH. Though the exact significance of this observation is not clear, we speculate that the melanocytic hyperplasia could be related to an inductive phenomenon or constitute a part of the overall hamartomatous process. Careful attention to epidermal changes in future cases of RMH may help in understanding the significance of this association and refine our knowledge of the pathogenesis of RMH.

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Congenital smooth muscle hamartoma. A report of six cases and a review of the literature

Cited by 40 publications

References 14 publications

Smooth muscle tumors of soft tissue and non-uterine viscera: biology and prognosis

Smooth muscle tumors of soft tissue and non-uterine viscera: biology and prognosis

An unusual hamartoma of the folliculosebaceous‐apocrine unit: a case report

Rhabdomyomatous Mesenchymal Hamartoma With Lentiginous Melanocytic Hyperplasia: An Inductive Phenomenon or Component of the Hamartoma?

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