Juvenile Hyalin Fibromatosis

Kitano, Yukio

doi:10.1001/archderm.1976.01630250050016

Cited by 60 publications

(44 citation statements)

References 4 publications

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“…It has been shown previously that cAMP stimulates DNA synthesis by human melanocytes in vitro (28) and that human melanocytes can replicate in vitro if the medium contains cholera toxin and isobutylmethylxanthine (29). Moreover, Pawelek et al (30) have shown that some variants of Cloudman S91 melanoma require cAMP to grow at the rate normally observed in wild-type cells.…”

Section: Discussionmentioning

confidence: 97%

Selective proliferation of normal human melanocytes in vitro in the presence of phorbol ester and cholera toxin.

Eisinger¹,

Marko²

1982

Proc. Natl. Acad. Sci. U.S.A.

483

256

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Cultures consisting almost entirely of human melanocytes were obtained from epidermal single-cell suspensions by using phorbol 12-myristate 13-acetate (10 ng/ml) in the culture medium. At this concentration, phorbol ester is toxic to human keratinocytes but not to melanocytes. When the seeding density was optimal (0.8-2 x 104/cm2) and the medium contained both phorbol ester and cholera toxin, melanocytes proliferated extensively. Under these conditions, human melanocytes could be passaged serially in vitro and grown in quantity. This cell culture system can thus be used to answer basic questions related to pigment cell biology and may serve as a control for studies of malignant melanocytes.Melanocytes constitute a minor component of the cell population in normal epidermis, being scattered at relatively low density throughout the entire epidermal sheet. The keratinocytes/ melanocytes ratio is -=35:1 (1). In normal epidermis, melanocytes undergo very little replication because they are rarely shed; on the other hand, keratinocytes undergo replication to replace those that are shed (2-4). In vitro studies ofmelanocytes have therefore had to overcome the following difficulties: (i) the lack of a good source tissue that contains melanocytes as the major component; (ii) the slow division rate ofmelanocytes; and (iii) the presence ofcells that grow faster than melanocytes, such as keratinocytes and fibroblasts.Current cell-culture techniques yield heterogeneous cell populations and lead to eventual overgrowth by keratinocytes and fibroblasts (for review, see ref. 5). Thus, studies of in vitro characteristics of human melanocytes have been complicated by the presence of contaminating populations of rapidly multiplying cells other than melanocytes. This has prevented subculturing and long-term study of melanocytes in culture.This paper presents evidence that, by using phorbol 12-myristate 13-acetate (PMA) (10 ng/ml), selective plating of melanocytes from a mixed cell population can be achieved. Moreover, in addition to suppressing the growth of keratinocytes, PMA also promotes the growth of melanocytes. By using a combination of PMA with cholera toxin, extended growth of melanocytes can be obtained. MATERIALS AND METHODSTissue Culture. Epidermal single-cell suspensions were prepared as described (6). Briefly, facial skin was reduced to split skin thickness (15/1000 in.; 1 in. = 2.54 cm) using a hand dermatome with a disposable blade (DAVOL, Cranston, RI); foreskin samples were freed from fatty tissue and washed with medium containing antibiotics. Each sample was cut into 2 x 5 mm pieces and washed in 0.02% EDTA (Sigma), and 0.5-1.5 g of tissue was placed in 2.5 ml of 0.25% trypsin (diluted 1:250; Difco) at 40C for 12-15 hr. Then, the trypsin was replaced by growth medium [Eagle's minimal essential medium with Earle's salts/0.01 mM nonessential amino acids (GIBCO)/2 mM L-glutamine (GIBCO)/5% fetal calf serum, pH 7.2, containing penicillin at 100 units/ml, streptomycin at 0.1 mg/ml, and Fungizone (GIBCO) at 0.25 ,...

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Section: Discussionmentioning

confidence: 97%

Selective proliferation of normal human melanocytes in vitro in the presence of phorbol ester and cholera toxin.

Eisinger¹,

Marko²

1982

Proc. Natl. Acad. Sci. U.S.A.

483

256

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“…Especially, roentogenographic study of bones reveal numerous lesions, which are clearly different from those of well-known genetic mucopolysaccharidoses or I-cell disease (Pureti6 et aI., 1962;Ishikawa et al, 1964Ishikawa et al, , 1973Horio et al, 1968;Gutidrrez et al, 1973;Kitano, 1976;Gianotti et al, 1977, present case).…”

Section: Discussionmentioning

confidence: 52%

“…There was evidence for familial occurrence in 11 cases from 5 families, showing a horizontal appearance. The segregation ratio in 16 cases, except for 2 cases (Kitano, 1976;Gianotti et al, 1977), was 0.1786__0.0726. These findings strongly suggest that this disorder is due to an autosornal recessive gene in agreement with Witkop (1971), Kitano et al (1972) and Ishikawa et al (1973).…”

Section: Discussionmentioning

confidence: 89%

“…As to diagnosis, well-known genetic mucopolysaccharidoses and I-cell disease can be excluded by careful examination of clinical manifestations, roentogenographic findings of bones, histological findings of the lesion and cytological findings of cultured cells (Kitano et al, , 1976. Especially, roentogenographic study of bones reveal numerous lesions, which are clearly different from those of well-known genetic mucopolysaccharidoses or I-cell disease (Pureti6 et aI., 1962;Ishikawa et al, 1964Ishikawa et al, , 1973Horio et al, 1968;Gutidrrez et al, 1973;Kitano, 1976;Gianotti et al, 1977, present case).…”

Section: Discussionmentioning

confidence: 99%

“…Costa et al, 1975; juvenile hyalin fibromatosis. Kitano, 1976; fibromatose hyaline juv6nile. Gianotti et al, 1977).…”

Section: Introductionmentioning

confidence: 99%

See 2 more Smart Citations

Puretic syndrome-gingival fibromatosis with hyaline fibromas

et al. 1980

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Fibromatosis hyalinica multiplex (juvenile hyalin fibromatosis) light microscopic, electron microscopic, immunohistochemical, and biochemical findings

Remberger¹,

Krieg

Kunze³

et al. 1985

Cancer

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Fibromatosis hyalinica multiplex juvenilis (juvenile hyalin fibromatosis) is a very rare mesenchymal dysplasia, probably inherited as an autosomal-recessive trait. Two nonrelated cases are reported. Among the clinical features, the most impressive lesions are multiple slowly growing subcutaneous nodules, hypertrophic gingiva, flexural contractures with joint stiffness and radiolucent bone destructions. Light microscopic examination of the nodules reveals tumor-like deposits of an amorphous hyaline ground substance with delicate staining properties situated partly between cellular and vascular areas. Ultrastructural characteristics are cystic, dilated rough endoplasmatic reticulum and cystic Golgi vesicles which contain a fine fibrillar material that is also found in the ground substance. Immunohistochemical examination shows collagen type I and type III in the hyaline material, but not type II and type IV. Quantitative biochemical investigation reveals a normal ratio of collagen types I and III.

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Juvenile Hyalin Fibromatosis

Cited by 60 publications

References 4 publications

Selective proliferation of normal human melanocytes in vitro in the presence of phorbol ester and cholera toxin.

Selective proliferation of normal human melanocytes in vitro in the presence of phorbol ester and cholera toxin.

Puretic syndrome-gingival fibromatosis with hyaline fibromas

Fibromatosis hyalinica multiplex (juvenile hyalin fibromatosis) light microscopic, electron microscopic, immunohistochemical, and biochemical findings

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