Acquired smooth muscle hamartoma with sebaceous component / Hamartoma de músculo liso adquirido con componente sebáceo

Abstract Background: Acquired smooth muscle hamartoma (ASMH) is a rare benign lesion characterized clinically by hyperpigmented plaques with hypertrichosis and some follicular papules. The main histologic finding is the presence of disorganized smooth muscle bundles in the dermis. Only 25 cases of ASMH have been reported in the literature. Clinical case: We present the case of an 18-year-old male who reported a pigmented area and increased hair growth on the left hemifacial with one year of evolution. Clinically, a plaque was observed in the preauricular region and on the left cheek with a linear Blaschkoid path, consisting of hyperpigmentation, hypertrichosis, and some papular lesions, with negative pseudo-Darier sign. Histological analysis showed an increase in the number of smooth muscle bundles in the middle and deep dermis surrounding abundant sebaceous glands and numerous hair follicles in different stages of evolution. Conclusions: The sebaceous component in this lesion was prominent. Therefore, we considered this lesion part of a spectrum where the acquired smooth muscle hamartoma and folliculosebaceous cystic hamartoma are found at the extremes. This case would fall in the middle of the range, as it combines both histological features.

Resumen Introducción: El hamartoma de músculo liso adquirido (HMLA) es una lesión benigna adquirida, poco frecuente, caracterizada clínicamente por presentar placas hiperpigmentadas, con hipertricosis y algunas pápulas foliculares. El principal hallazgo histológico es la presencia de abundantes haces de músculo liso desorganizados en la dermis. Se han reportado solo 25 casos de HMLA en la literatura. Caso clínico: Se presenta el caso de un paciente de sexo masculino de 18 años que refirió una zona pigmentada y el aumento de vello en la hemicara izquierda con un año de evolución. Clínicamente se observó una placa en la región preauricular y mejilla izquierda con trayecto lineal blaschkoide, constituida por hiperpigmentación, hipertricosis y algunas lesiones papulares, con signo pseudo-Darier negativo. Histológicamente se encontró un aumento en el número de haces de músculo liso en la dermis media y profunda rodeando abundantes glándulas sebáceas, así como numerosos folículos pilosos en diferentes estadios de evolución. Conclusiones: El componente sebáceo en esta lesión fue muy marcado, por lo que se considera que forma parte de un espectro donde en los extremos se encuentran el hamartoma de músculo liso adquirido y el hamartoma quístico folículo sebáceo. El presente caso se encontraría en medio, ya que combina ambas características histológicas.

Hamartoma mioide, serie de casos y revisión de la literatura / Myoid hamartoma: a case series and literature review

Abstract: Myoid hamartoma is a rare entity, and was first described by Davies and Riddell in 1973. It is pathologically defined as the mixture of fatty glandular tissue and fibrous connective tissue, associated with a widespread focus of fusiform muscle cells. Six cases of HM diagnosed in seven patients are presented, with emphasis on imaging findings and characteristics of their respective core biopsies. The importance of biopsy in these lesions is required to differentiate malignant diseases. There is no need for excisional biopsy, because this is not associated with high-risk lesions or carcinomas.

Resumen: El hamartoma mioide es una lesión infrecuente; fue descrita por primera vez por Davies y Riddell en el año 1973; se define histológicamente como la mezcla de tejido glandular adiposo y conjuntivo fibroso, asociado a extensos focos de células musculares fusiformes. Se presentan 7 casos de HM diagnosticados en 7 pacientes, haciendo énfasis en las características imagenológicas y los hallazgos de las respectivas biopsias core. La importancia de realizar biopsia en estas lesiones radica en la necesidad de diferenciarla de patologías malignas; así mismo no existe necesidad de biopsia excisional, ya que se trata de una entidad que no se asocia a lesiones de alto riesgo o con cáncer.

Congenital Smooth Muscle Hamartoma with Follicular Spotted Appearance / 대한피부과학회지

A congenital smooth muscle hamartoma is a rare benign cutaneous abnormality and histologically characterized by smooth muscle hyperplasia with a great number of long and straight bundles of smooth muscle at various angles of the orientation, throughout the dermis. A congenital smooth muscle hamartoma usually presents as a congenital hairy patch or plaque with or without hyperpigmentation on the trunk or extremities. The congenital smooth muscle hamartoma with follicular spotted appearance is a rare clinical variant, and is appeared as a patch with marked follicular papules. Herein, we report a 16-year-old girl with this rare patch follicular variant of congenital smooth muscle hamartoma, on the left upper arm.

A Case of Acquired Smooth Muscle Hamartoma on the Sole

A smooth muscle hamartoma is a benign proliferation of smooth muscle bundles within the dermis. It arises from smooth muscle cells that are located in arrector pili muscles, dartos muscles, vascular smooth muscles, muscularis mammillae and the areolae. Acquired smooth muscle hamartoma (ASMH) is rare, with only 10 such cases having been reported in the English medical literature to date. Most of these cases of ASMH were shown to have originated from arrector pili and dartos muscles. Only one case was reported to have originated from vascular smooth muscle cells. A 21 year-old woman presented with a tender pigmented nodule, with numbness, on the sole of her foot, and this lesion had developed over the previous 18 months. The lesion showed no hyperpigmentation or hypertrichosis, and the biopsies demonstrated increased smooth muscle bundles in the dermis, and especially around the blood vessels. Moreover, the specimens stained positive with Masson trichrome stain and alpha-smooth-muscle actin antibodies, thus supporting our diagnosis of ASMH of the foot sole. Herein, we report on a rare case of ASMH on the foot sole, and this lesion originated from vascular smooth muscle cells. This type of case has not been previously described in the English medical literature.

A Case of Congenital Smooth Muscle Hamartoma Presenting as a Human Tail / 대한피부과학회지

A human tail is a rare congenital anomaly, and is characterised by a prominent lesion from the lumbosacrococcygeal region. Human tails are classified into 'true tails' and 'pseudotails'. True tails are comprised of only mesenchymal tissue. They are presumed to be remnants of the embryologic tail. All other lumbosacrococcygeal protrusions are summarized as pseudotails. Congenital smooth muscle hamartoma usually presents as a well-circumscribed, hypertrichotic, hyperpigmented or skin-colored patch or plaque on the trunk or an extremity at birth. Histologically, numerous thick, long, well-defined bundles of smooth muscle fibers are scattered throughout the dermis in various directions. We report a case of congenital smooth muscle hamartoma in a 15-year-old male patient who presented with a tail-like protruding mass accompanied by hypertrichosis and underlying soft tissue deviation.

Three Cases of Congenital Smooth Muscle Hamartomas

Congenital smooth muscle hamartomas (CSMHs) usually appear at birth as skin-colored or slightly hyperpigmented patches or plaques on the trunk or extremities and are often covered by and increased amount of vellus hair. Histopathologically, it represents a proliferation of smooth muscle bundles within the dermis(1,2). We herein report on three cases of localized CSMHs; Case 1 was a 2-month-old boy who presented with a skin-colored, 6x5cm-sized plaque with folds, hypertrichosis and pseudo- Darier's sign on the face, an unusual site. But there was no hyperpigmentation. Case 2 and 3 were both 1-month-old boys who presented with typical hyperpigmented, hairy plaques on the lateral aspect of the left thigh.

A Case of Aquired Smooth Muscle Hamartoma with Follicular Spotted Appearance / 대한피부과학회지

Smooth muscle hamartoma is a benign hamartomatous condition, presenting clinically as a skin-colored or hyperpigmented patch or plaque and histologically as dermal smooth muscle hyperplasia. It is thought that Becker's nevus and smooth muscle hamartoma are distinct entities at either end of a spectrum. It can either be congenital or acquired We herein report a case of acquired smooth muscle hamartoma with follicular spotted appearance. We consider it as a rare clinical variant of smooth muscle hamartoma, which is clearly different from Becker's nevus.

A Case of Acquired Smooth Muscle Hamartoma on the Sole / 대한피부과학회지

Smooth muscle hamartoma (SMH) is a benign proliferation of smooth muscle bundles within the dermis. It arises from either the arrector pili muscles, dartos muscles, smooth muscle cells in blood vessels and lymphatics, muscularis mamillae and areola, or cutaneous adnexa. Acquired smooth muscle hamartoma is very rare, and only 12 cases have been published to date. All reported cases are originated from arrector pili or dartos muscles. Herein, we describe a case of acquired smooth muscle hamartoma which originated from the smooth muscle cells in blood vessels of the right sole. To our knowledge, this is the first reported case which has originated from the smooth muscle cells in blood vessels.

A Case of Congenital Smooth Muscle Hamartoma / 대한피부과학회지

Congenital smooth muscle hamartoma usually presents as a well-circumscribed, hypertrichotic, hyperpigmented or skin colored patch or plaque on the trunk or an extremity at birth. Histologically, numerous thick, long, well defined bundles of smooth muscle fibers are scattered throughout the dermis in various directions. We report a case of congenital smooth muscle hamartoma in an 8-month-old female infant who showed hypertrichotic, skin colored plaque on the lumbosacral area since at birth. The pseudo-Darier sign was positive. Histopathologic findings were numerous, well-defined bundles of smooth muscle fibers in the dermis (H and E, Masson-trichrome stain).

A Case of Acquired Cutaneous Smooth Muscle Hamartoma

Smooth muscle hamartoma was first described by Stokes in 1923 as the disease characterized by increase of well-defined bundles of smooth muscle fibers in the dermis. It can be either congenital or acquired. Acquired ones are aften in association with Becker's nevus. We present a case of acquired smooth muscle hamartoma that did not show any pigmentation or hair growth.

Congenital Smooth Muscle Hamartoma: a Patchy Follicular Variant

Cutaneous smooth muscle hamartomas are benign proliferations of smooth muscle bundles within the dermis. They can be congenital or acquired, and most cases are congenital. Congenital smooth muscle hamartomas (CSMHs) usually manifest at birth as well-circumscribed, frequently hypertrichotic, hyperpigmented or skin-colored patches or plaques on trunk or extremities. We report a case of CSMH in a 10 year-old girl, who showed a localized skin-colored patch showing prominent follicular papules on the lateral aspect of her right upper arm, which were found at birth. There was no hypertrichosis and the pseudo-Darier sign was negative. This patchy follicular variant is the less common clinical type of the disease.

A Case of Congenital Smooth Muscle Hamartoma / 대한피부과학회지

Congenital smooth muscle hamartoma(CSMH), a rare congenital benign tumor of the skin, usually appears at birth as skin colored or slightly hyperpigmented patches or plaques, sometimes with lightly pigmented vellus hairs. Histopathologically it has a marked hyperplasia of well-demarcated smooth muscle bundles in the dermis. We report a case of CSMH in a 4-month-old male infant, who had had hypertrichosis, rippling and multiple light brown colored macules on both his lower extremites since birth. Histopathologically, markedly proliferated smooth muscle buildles were scattered throughout the retucular dermis.

A case of smooth muscle hamartoma associated with Becker's nevus / 대한피부과학회지

We report herein a case of smooth muscle hamartoma associated with Beckers nevus in a 5-year-old female. The child presented with a slight.ly brownish. 2 x 3 cm sized, pigmentecl patch with follicular accentuation on the right forearm, witch had been observecl since age of 2. Histopathologic findings showed the epidermal changes most consistent with Becker's nevus and the presence of irregularly arranged hyperplastic smooth muscle hundless in the dermis.

A Case of Becker's Nevus Associated with Smooth Muscle Hamartoma / 대한피부과학회지

We report a case of Becker's nevus associated with smooth muscle hamartoma, in a 21-year-old male patient, which shows clinically match-head sized, flat topped, round to oval, grouped papules with hairs on the outer surface of the right arm, and microscopically reveals numerous bundles of smooth muscle fiber in the dermis.

A Case of Congenital Smooth Muscle Hamartoma / 대한피부과학회지

Congenital smooth musele hamartomas appear at birth as hypertrichotic patches or plaques with or without hyperpigmentation. Histologic characteristic is hyperplasia of dermal smooth muscle bundles. We report a case of congenital smooth muscle harnartoma in 5-year-old female, who showed localized excessive-hairy, skin-colored patch on the lateral surface of left. elbow, which were found at birth. Biopsy specimen showed hyperpigmentation of the basal layers of epidermis, melanophages in upper dermis, and hyperplasia of smooth muscle bundles in lower dermis.

A Case of Chondrodermatitis Nodularis Chronica Helicis / 대한피부과학회지

A 51-year-old man had small, tender, skin colored firm nodule on the helix of the both ears for eight months. He was found to have classic features of chondrodermatitis nodularis chronica helicis, namely, tender nodule less than 1 cm in length with central crusting, localization to helix, age, men, and histologic findings. The histologic examination of the biopsy specimen showed the presence of thinned epiderrnis on center. acanthosis on the adjacent epidermis, with hyperkeratosis and parakeratosis, and cleft between epidermis and dermis. In the dermis, highly vascularized connective tissue and chronic inflarnmatory infiltrate were visible with perichondrial fibrosis. Elastic tissue stain showed degeneration of elastic fibers. After complete excision, no recurrence was obsered until now.