Diagnosis and surgical treatment of dysplasia epiphysealis hemimelica. A report of nine cases

Bosch, C.; Assi, Chahine; Louahem, Djamel; Alkar, Fanny; Mazeau, Perrine Coste; Delfour, Christophe; Canavese, Federico; Prodhomme, O.; Cottalorda, J.

doi:10.1016/j.otsr.2014.07.022

Cited by 16 publications

(22 citation statements)

References 22 publications

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“…13-21 The aim of this study was to histologically compare DEH and osteochondromas and investigate whether both diseases are histologically identical diseases.…”

Section: Discussionmentioning

confidence: 99%

Dysplasia epiphysealis hemimelica: A histological comparative study with osteochondromas

Stevens

Welting

Witlox

et al. 2017

Journal of Children's Orthopaedics

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PurposeDysplasia epiphysealis hemimelica (DEH) is a rare developmental disorder resulting in epiphyseal overgrowth. Based on histological appearance, it is often described as an osteochondroma or osteochondroma-like lesion, although clinical differences exist between DEH and osteochondromas. The aim of this study was to test whether DEH and osteochondromas are histologically identical diseases.MethodsTissue samples of two age- and gender-matched cases with DEH and hereditary multiple exostoses were histologically compared. Sections were stained with Safranin-O for detection of proteoglycans and immunohistochemistry was performed for detection of collagen type II, collagen type X as a marker of hypertrophic chondrocytes and Sox9 as a marker of proliferative chondrocytes. Due to the rarity, descriptions of the included DEH patients were outlined.ResultsHistologically, chondrocyte clusters in a fibrillary matrix, a thick disorganised cartilage cap and ossification centres with small amounts of unabsorbed cartilage, were observed in DEH. In contrast, cartilage organisation of osteochondromas displays characteristics of the normal growth plate. Collagen type II was clearly detected in the cartilaginous extracellular matrix in osteochondromas, while weak expression was observed in DEH. Collagen type X was not detected in DEH, while expressed in the matrix surrounding hypertrophic chondrocytes in osteochondromas. Sox9 staining was positive in hypertrophic chondrocytes in osteochondromas, while expressed in nuclei of chondrocyte clusters in DEH.ConclusionBoth morphological and immunohistological differences were observed in histological sections of DEH and osteochondromas. These results support the previously identified clinical, radiological and genetic differences and imply a different aetiology between DEH and osteochondroma formation.

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“…13-21 The aim of this study was to histologically compare DEH and osteochondromas and investigate whether both diseases are histologically identical diseases.…”

Section: Discussionmentioning

confidence: 99%

Dysplasia epiphysealis hemimelica: A histological comparative study with osteochondromas

Stevens

Welting

Witlox

et al. 2017

Journal of Children's Orthopaedics

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“…Reports of outcomes after excision of intra-articular lesions vary. Kuo et al 4 reported a high rate of complications after excision of intra-articular lesions, whereas Bosch et al 16 reported excellent results with early intervention. The only clear indication for excision of intra-articular lesions is the presence of symptomatic loose bodies.…”

Section: Discussionmentioning

confidence: 99%

Trevor's Disease: Management Difficulties and Proposed Classification

Clarke¹

2016

Orthopedics

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Dysplasia epiphysealis hemimelica, also known as Trevor's disease, is a rare developmental disorder with osteocartilagenous overgrowth of the epiphysis or epiphyseal equivalent. The condition bears similarities to osteochondroma in terms of its radiographic appearance, but differs in its pathobiology and geographic occurrence. Unlike the metaphyseal occurrence of osteochondromata, it arises from the epiphysis. The clinical presentation is wide and varied, but mechanical symptoms and deformities predominate. Early reports of the condition suggested involvement of the lower limbs only. However, since then, numerous reports have indicated a more generalized distribution. Difficulties in management and recurrence rates seem to hinge on whether its origin is intra-articular or extra-articular. A new classification system to include these parameters is discussed. [Orthopedics.2016; 39(5):e967-e969.].

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“…Dysplasia epiphysealis hemimelica (DEH) is a rare asymmetrical epiphyseal cartilaginous overgrowth that is commonly seen in children or teenagers, mostly males [ 1 ]. It was originally described as a “tarsomegalie” in 1926 by Mouchet and Berlot [ 2 ].…”

Section: Introductionmentioning

confidence: 99%

“…The medial side is more common. The lower limbs are commonly affected, whereas the upper limbs and spine are rare sites [ 1 ], and dysplasia usually occurs as a single bone protuberance at the epiphysis [ 6 ]. We report a case of talus DEH accompanied by multiple free bodies in a 7-year-old patient with ankle instability, which resulted in recurrence after primary free body removal and microfracture technique to the talus lesion and obtained successful short-term results using secondary arthroscopic removal with lateral ankle ligament repair.…”

Section: Introductionmentioning

confidence: 99%

Juvenile Dysplasia Epiphysealis Hemimelica with Multiple Ankle Free Body

Sato

Chang

Kasai

et al. 2021

Case Reports in Orthopedics

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Dysplasia epiphysealis hemimelica (DEH), also known as Trevor’s disease, is a rare overgrowth of cartilage that commonly arises in the epiphyseal bone of children. We report a rare case of DEH originating from a talus accompanied by multiple intra-articular free bodies in a 7-year-old patient with ankle instability. After the primary surgery for free body removal and microfracture technique for the cartilage defects in the ankle joint, the free body recurred. Secondary surgery of arthroscopic free body removal with lateral ankle ligament repair succeeded in treating the patient, without further recurrence of the free body.

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Diagnosis and surgical treatment of dysplasia epiphysealis hemimelica. A report of nine cases

Cited by 16 publications

References 22 publications

Dysplasia epiphysealis hemimelica: A histological comparative study with osteochondromas

Dysplasia epiphysealis hemimelica: A histological comparative study with osteochondromas

Trevor's Disease: Management Difficulties and Proposed Classification

Juvenile Dysplasia Epiphysealis Hemimelica with Multiple Ankle Free Body

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