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PMID: 987909 Published · ppublish English Case Reports Journal Article Research Support, U.S. Gov't, P.H.S.

Chondrodysplasia punctata--rhizomelic form. Pathologic and radiologic studies of three infants.

European journal of pediatrics ·Vol. 123 ·No. 2 ·1976-09-01 ·Pages 89-109

Gilbert EF, Opitz JM, Spranger JW, Langer LO, Wolfson JJ, Viseskul C

Abstract

Pathologic, ultrastructural and radiologic studies are described on 3 infants with the rhizomelic form of chondrodysplasia punctata. Radiologic criteria in the young infant include radiolucent coronal clefts dividing all or most of the thoracic and lumbar vertebral bodies, short humeri with flared metaphyses and punctate calcifications commonly present adjacent to the ossified ischial and pubic bones and less commonly in other locations. In late infancy and childhood the radiologic criteria include demineralization in all bones with slow maturation, flat vertebral bodies, short humeri and femora, metaphyseal flaring, especially in the distal humerus, proximal femur and proximal tibia, immature shape of pelvis, and disappearance of the punctate calcifications with advancing age. The histologic changes of the resting cartilage include areas of degenerating cartilage which had become partially calcified, cystic changes with severe disturbance of the maturation of the cartilage at the physial plate, and the formation of cancellous bone directly on resting cartilage. Ultrastructural changes are characterized by degeneration of chondrocytes, delicate collagenous fibrils without visible periodicity, and the presence of flocculent material within greatly distended endoplasmic reticulum.

MeSH Terms
Bone and Bones/pathology,ultrastructure Cartilage/pathology Cartilage, Articular/ultrastructure Cataract/complications Cerebral Cortex/pathology Child Child, Preschool Chondrodysplasia Punctata/pathology Contracture/complications Female Femur/pathology Humans Humerus/pathology Infant Infant, Newborn Infant, Newborn, Diseases/pathology Male Microcephaly/complications Neurons/ultrastructure Patella/pathology Psychomotor Disorders/complications Skin Diseases/complications
Authors & Affiliations
6 authors, click to expand affiliations / ORCID
Gilbert E F
Opitz J M
Spranger J W
Langer L O
Wolfson J J
Viseskul C
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28 references, click to expand
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Article Info
Journal
European journal of pediatrics
Abbr.
Eur J Pediatr
ISSN
0340-6199
Published
1976-09-01
Pages
89-109
Language
English
Region
Germany
NLM ID
7603873
Subset
IM
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