G. Nishimura et al., GENERALIZED METAPHYSEAL MODIFICATION WITH CONE-SHAPED EPIPHYSES FOLLOWING LONG-TERM ADMINISTRATION OF 13-CIS-RETINOIC ACID, European journal of pediatrics, 156(6), 1997, pp. 432-435
We report on a 6-year-old girl with short stature which developed foll
owing the administration of 13-cis-retinoic acid (a synthetic derivati
ve of vitamin A or retinoid) for 40 months as adjunct chemotherapy for
neuroblastoma. Radiographic examination suggested osteophyte formatio
n in the cervical spine, which is the most common skeletal manifestati
on of retinoid toxicity [10, 11]. In addition, severe metaphyseal cupp
ing with a cone-shaped epiphysis primarily affecting rapidly growing l
ong bones was found, which represented impaired enchondral ossificatio
n. This epi-metaphyseal alteration, though unusually severe, was remin
iscent of the premature epiphyseal closure which has been described as
an adverse effect of 13-cis-retinoic acid [10-12]. Other minor skelet
al changes included posterior scalloping of the vertebral bodies and i
ncreased interpediculate distances, which were related to a widened sp
inal canal found on CT. A literature search disclosed several primary
skeletal dysplasias with superficial radiological similarities to thos
e of the present patient. However, these entities showed significant c
linical and radiological differences from our patient. Conclusion The
precise cause of the generalized skeletal alteration in the present pa
tient remained unknown, but it conceivably resulted from the administr
ation of 13-cis-retinoid acid.