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At least 19 recordsLinked to original sources
PubMedDate not supplied

Respiratory epithelial adenomatoid hamartomas and chondroosseous respiratory epithelial hamartomas of the sinonasal tract: a case series and literature review.

BACKGROUND: Respiratory epithelial adenomatoid hamartomas (REAH) and chondroosseous respiratory epithelial (CORE) hamartomas are rare sinonasal/nasopharyngeal lesions first characterized in 1995. Although REAH and CORE hamartomas are benign, nonneoplastic lesions, awareness and recognition of these lesions are important because they can be confused grossly and microscopically with more threatening sinonasal tumors. METHODS: This report presents two cases of REAH and one case of a CORE hamartoma. The literature regarding REAH and CORE hamartomas is reviewed, and their distinction from related entities of the sinonasal tract and nasopharynx is discussed. CONCLUSIONS: Misdiagnosing REAH or CORE hamartoma as either inverted papilloma or adenocarcinoma may lead to far more aggressive surgical intervention then is necessary.

Aged, 80 and over↗
PubMed1998

Nasal chondromesenchymal hamartoma: an upper respiratory tract analogue of the chest wall mesenchymal hamartoma.

Nasal chondromesenchymal hamartoma is the suggested appellation for a tumefactive process of the nasal passages and contiguous paranasal sinuses in seven children with a detectable mass in the nose. With the exception of one patient who was 7 years of age at diagnosis, the others were 3 months of age or less upon recognition of the mass. Two children were diagnosed in the first 2 weeks of life. Imaging studies showed a complex solid and cystic mass or masses filling the nasal cavity and extending into the ethmoid sinuses in most cases. Erosion of the surrounding bone, including the cribriform plate, resulted in an intracranial component in the four cases. Surgical resection was the treatment of choice despite its technical difficulties that often necessitated a combined intranasal and intracranial approach. Residual disease with continued growth in one case was the clinical outcome in two children, and the remaining five patients have not experienced any further difficulties. The piecemeal fragments of tissue disclosed a collage of histologic features, but the basic morphologic elements were well-demarcated nodules of cartilage with some variation in the cellular density and maturation of the chondrocytes, a myxoid to spindle cell stroma, focal osteoclastlike giant cells in the stroma, and erythrocyte-filled spaces resembling those of the aneurysmal bone cyst. Two of the tumors were less polymorphous or complex in their spectrum of histologic features. These nasal masses have similarities to the so-called chest wall hamartoma or mesenchymal hamartoma of the chest wall in terms of the clinical presentation in infancy and the basic cartilaginous character of both entities. There is a degree of presumption in the designation of these nasal and chest wall tumors as hamartomas because the pathogenesis has not been established for either entity.

Actins↗
PubMed2004

Myoid hamartoma of the liver--a novel variant of hamartoma developing in the hilar region and imitating a malignant liver tumor.

BACKGROUND: We report here an unusual variant of hepatic mesenchymal hamartoma exhibiting a marked myoid differentiation and clinically imitating hilar malignancy. CASE REPORT: A 17-year-old patient was admitted to the hospital with the suspicion of Klatskin's tumor. Three months before he had presented with jaundice and light stools. Imaging techniques demonstrated a solid lesion (3 cm) situated in liver segment IV. Biochemical tests detected an increased level of bilirubin, alkaline phosphatase, AspAt, ALAT, and slightly increased CA19-9. Diagnosed with perihilar malignancy, the patient underwent left-sided hemihepatectomy with hepaticojejunostomy. The liver resection specimen showed an unencapsulated solid hilar tumor (5x3x3.5 cm), consisting of eosinophilic spindle-like cells with blunt-ended nuclei. Within the lesion we found numerous biliary ductules. Focally, numerous plasma cells and eosinophils were found, but no cystic spaces. There was no cellular atypia. Apart from the lesion the liver revealed intensified fibrosis of portal areas. Immunohistochemical studies demonstrated positivity of most spindle-like cells for SMActin, whereas only a few cells were CD34- and desmin-positive. Ki67 was positive in less than 5% cells. CONCLUSIONS: Given the indistinct border and heterogeneous morphology, with spindle-like myoid cells constituting most of the tissue, interspersed with biliary ductules, we suggest that this tumor is an unusual variant of mesenchymal hamartoma. As the mesenchymal component in this lesion was mainly represented by cells of muscular origin, we suggest classifying this lesion as an previously undescribed form of mesenchymal hamartoma, and propose the term, myoid hamartoma of the liver.

Adolescent↗
PubMed1985

Congenital arrector pili hamartoma. A case report and review of the spectrum of Becker's melanosis and pilar smooth-muscle hamartoma.

Congenital pigmented arrector pili hamartomas are unique malformations of epidermis and pilar apparatus usually appearing as localized, lightly pigmented, hairy plaques. Characteristic microscopic features include smooth-muscle proliferation similar to irregularly disposed arrectores pilorum, and slight elongation of epidermal rete with hypermelanosis of the basal unit. An otherwise normally developed child who had this hamartoma at birth is described in an attempt to clarify the relationship between pilar smooth-muscle hamartomas and Becker's melanosis. We propose that these two entities belong at different poles of the same developmental spectrum of hamartomatous change.

Diagnosis, Differential↗
PubMed1995

Dermal dendrocyte hamartoma with stubby white hair: a novel connective tissue hamartoma of infancy.

A previously undescribed hamartoma with stubby white hair was observed in a 1-week-old girl. A deep red, soft nodule with fine wrinkles was present on the back. White bizarre short thick hairs with irregular exterior cuticular squamae were noted. The main cells proliferating in the nodule were fibroblast-like spindle cells that had dendrites and showed positive staining for CD34 antigen. These cells surrounded vessels and nerves. In addition, there were immature hair follicles, relatively thick-walled small vessels, and small adipose cells with fine connective tissue. This hamartoma was considered to be a dermal dendritic cell hamartoma originating from CD34-positive cells.

Adipose Tissue↗
PubMed1993

Cutaneous hamartoma of adnexa and mesenchyme. A variant of folliculosebaceous cystic hamartoma with vascular-mesenchymal overgrowth.

We describe a hamartoma with folliculosebaceous and mesenchymal components. Striking vascular-mesenchymal elements distinguish this entity from previously described hamartomas and other adnexal proliferations. It is also distinct from the recently described folliculosebaceous cystic hamartoma but likely represents a variant with mesenchymal overgrowth.

Cysts↗
PubMed1996

Autosomal dominant primary hyperparathyroidism and jaw tumor syndrome associated with renal hamartomas and cystic kidney disease: linkage to 1q21-q32 and loss of the wild type allele in renal hamartomas.

Hereditary hyperparathyroidism-jaw tumor syndrome (HPT-JT) is an autosomal dominant disease (OMIM 145001) that has recently been mapped to chromosomal region 1q21-q32 (HRPT2). Here we report two families with HPT-JT syndrome in which adult renal hamartomas or cystic kidney disease were prominent associated features, possibly representing a new phenotypic variant of the HPT-JT syndrome. In the first family, renal lesions were present in five out of six affected individuals, whereas HPT and JT were seen in four and two cases, respectively. In the second family, JT was found in three of the five affected individuals and two affected members also exhibited polycystic kidney disease. The possibility of the latter cosegregating as a separate autosomal dominant gene can not be ruled out. A sex-dependent penetrance of primary HPT, resulting in predominantly male-affected cases was evident in the two families. Twenty microsatellite markers in the HRPT2 region were typed, in addition to markers in the multiple endocrine neoplasia (MEN) types 1 and 2 regions at 11q13 and 10q11. The disease in these two kindreds was linked to five markers in the 1q21-q32 region (logarithm-of-odds scores: 3.2-4.2), whereas linkage to the MEN1 and MEN2 regions was excluded. Meiotic recombinations detected in affected individuals placed the locus telomeric of D1S215, thus narrowing the HRPT2 region from > 60 to approximately 34 centimorgans. Loss of heterozygosity was studied in seven renal hamartomas from two affected individuals in the first family, as well as in a jaw tumor and a parathyroid tumor from the second family. All renal hamartomas showed loss of heterozygosity at the 1q21-q32 region. The losses invariably involved the wild type allele derived from the unaffected parent, suggesting the inactivation of a tumor suppressor gene in this region.

Adult↗
PubMed1979

Pleomorphic pulmonary hamartoma: an apparently unique variant of pulmonary hamartoma.

Pulmonary hamartomas of limited variety have been described. Most present as asymptomatic coin lesions in adults and consist of mesenchymal tissue, usually cartilage, in combination with irregular spaces lined by epithelium. Another form is found in the neonate and involves large portions or all of a lung. This is associated with a developmental aberration and is best described as a "congenital adenomatoid malformation". An apparently unique noninvasive tumor mass was resected from the lung of a middle aged man where it was associated with anomalous lung segmentation and the bronchial and blood supply to the lung. The tumor appeared to be undifferentiated by light microscopic criteria. Neurilemoma, leiomyoma, chordoid tumor, mixed tumor, and neurogenic sarcoma all entered the differential diagnosis. Ultrastructural examination demonstrated a highly complex and unique organization. Such a lesion, apparently developmental in this case, should be recognized and carefully distinguished from the malignant mesenchymal, neurogenic, and teratomatous lesions with which it may be confused. Electron microscopy may be helpful in this regard.

Adult↗