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Tissue chondrification and ossification in keloids with primary report of five cases.

ABSTRACT: Keloid is commonly regarded as a benign skin tumour. Some keloids clinically exhibit hard tissue texture similar to that of cartilage or bone. We hypothesized that the keloid pathological niche environment is likely to induce keloid MSCs towards chondrogenic or osteogenic differentiation and leads to cartilage or bone-like tissue formation. The differences in tissue ossification, histology, mechanical properties, abnormal extracellular matrices and chondrogenic/osteogenic gene expression among sclerous keloids (SKs), regular keloids (RKs) and normal skins (NKs) were carefully examined. The sporadic ossified islets existed in SK group whereas no ossified/chondrified islet was found in other groups by micro-CT reconstruction. H&E, Masson trichrome and safranin O staining revealed lacuna-like structures in SKs, which were featured as bone/cartilage histology. Immunohistochemical staining showed overproduction of osteoprotegerin, type I and III collagen in SK group but similar production level of aggrecan among three groups. The biomechanical analysis demonstrated the weakest compliance of SK tissues. In addition, SK fibroblasts exhibited a relatively slower proliferation rate but higher expression levels of osteogenic and chondrogenic genes among all three groups. These cell populations also showed the strongest potential for lineage transformation. In conclusion, we first reported the presence of ossified and chondrified matrices in some extremely hard keloids in the present study.

SUBMITTER: Li Q

PROVIDER: S-EPMC9615288 | biostudies-literature | 2022 Nov

REPOSITORIES: biostudies-literature

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Tissue chondrification and ossification in keloids with primary report of five cases.

Li Qiannan Q Tu Tian T Wu Xiaoli X Wang Wenbo W Gao Zhen Z Liu Wei W

International wound journal 20220321 7

Keloid is commonly regarded as a benign skin tumour. Some keloids clinically exhibit hard tissue texture similar to that of cartilage or bone. We hypothesized that the keloid pathological niche environment is likely to induce keloid MSCs towards chondrogenic or osteogenic differentiation and leads to cartilage or bone-like tissue formation. The differences in tissue ossification, histology, mechanical properties, abnormal extracellular matrices and chondrogenic/osteogenic gene expression among s ...[more]

PMID: 35315582

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Project description:BACKGROUND:Cutis tricolor is a skin abnormality consisting in a combination of congenital hyper- and hypopigmented skin lesions (in the form of paired macules, patches or streaks) in close proximity to each other in a background of normal skin. It is currently regarded as a twin-spotting (mosaic) phenomenon and today is clear that not all cases of cutis tricolor represent one single entity. This phenomenon has been reported so far either: (I) as an purely cutaneous trait; (II) as a part of a complex malformation phenotype (Ruggieri-Happle syndrome, RHS) including distinct facial features, eye (cataract), skeletal (skull and vertebral defects, and long bones dysplasia), nervous system (corpus callosum, cerebellar and white matter anomalies, cavum vergae and holoprosencephaly) and systemic abnormalities; (III) as a distinct type with multiple, disseminated smaller skin macules (cutis tricolor parvimaculata); and (IV) in association with other skin disturbances [e.g., cutis marmorata telangectasica congenita (phacomatosis achromico-melano-marmorata)] or in the context of other skin (e.g., ataxia-telangiectasia and phacomatosis pigmentovascularis, PPV) or complex malformation phenotypes (e.g., microcephaly and dwarfism). METHODS:(I) Review of the existing literature; and (II) information on our personal experience (clinical, laboratory and imaging data) on new cases with cutis tricolor seen and followed-up at our institutions during years 2010-2016. RESULTS:The existing literature revealed 19 previous studies (35 cases) with pure cutaneous or syndromic cutis tricolor phenomena. Our personal experience included 5 unpublished patients (3 boys; 2 girls; currently aged 2 to 14 years) seen and followed-up at our Institutions in Italy who had: (I) skin manifestations of the cutis tricolor type (N=5); (II) skeletal abnormalities including small skull (n=2), obtuse angle of mandible (n=3), mild to moderate scoliosis (n=3), vertebral defects (n=3), and long bones bowing (n=3); mild psychomotor delay (n=3); epilepsy (n=2); anomalies of the corpus callosum (n=3); and cavum vergae (n =2). CONCLUSIONS:This study further confirms and expands the overall phenotype of cutis tricolor. By literature review and personal experience we conclude that the skin abnormalities of the cutis tricolor type are stable over time; the skeletal defects are mild to moderate and do not progress or cause relevant orthopaedic complications; the neurological/behavioural phenotype does not progress and the paroxysmal events (when present) tend to decrease over time; there is a typical facial phenotype in some patients (long, elongated face, thick and brushy eyebrows, hypertelorism, deep nasal bridge with large bulbous nose and anteverted nostrils), which characterizes a somewhat distinct syndromic phenotype; some patients may develop early onset cataracts. The allelic dydymotic hypothesis of post-zygotic mutations likely involving the same gene loci could well explain the overall skin, bone, lens and nervous system phenomena of migration of different streaks of clones in the different tissues.

Dataset Information

Tissue chondrification and ossification in keloids with primary report of five cases.

Publications

Tissue chondrification and ossification in keloids with primary report of five cases.

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