European Journal of Pediatrics

, Volume 156, Issue 6, pp 432–435

Generalized metaphyseal modification with cone-shaped epiphyses following long-term administration of 13-cis-retinoic acid

  • G. Nishimura
  • H. Mugishima
  • J. Hirao
  • M. Yamato
GROWTH/DEVELOPMENT

DOI: 10.1007/s004310050631

Cite this article as:
Nishimura, G., Mugishima, H., Hirao, J. et al. Eur J Pediatr (1997) 156: 432. doi:10.1007/s004310050631

Abstract

We report on a 6-year-old girl with short stature which developed following the administration of 13-cis-retinoic acid (a synthetic derivative of vitamin A or retinoid) for 40 months as adjunct chemotherapy for neuroblastoma. Radiographic examination suggested osteophyte formation in the cervical spine, which is the most common skeletal manifestation of retinoid toxicity [10, 11]. In addition, severe metaphyseal cupping with a cone-shaped epiphysis primarily affecting rapidly growing long bones was found, which represented impaired enchondral ossification. This epi-metaphyseal alteration, though unusually severe, was reminiscent of the premature epiphyseal closure which has been described as an adverse effect of 13-cis-retinoic acid [10–12]. Other minor skeletal changes included posterior scalloping of the vertebral bodies and increased interpediculate distances, which were related to a widened spinal canal found on CT. A literature search disclosed several primary skeletal dysplasias with superficial radiological similarities to those of the present patient. However, these entities showed significant clinical and radiological differences from our patient.

Conclusion The precise cause of the generalized skeletal alteration in the present patient remained unknown, but it conceivably resulted from the administration of 13-cis-retinoid acid.

Key words 13-cis-retinoic acid  Neuroblastoma Metaphyseal cupping  Cone-shaped epiphysis  Short stature

Copyright information

© Springer-Verlag Berlin Heidelberg 1997

Authors and Affiliations

  • G. Nishimura
    • 1
  • H. Mugishima
    • 2
  • J. Hirao
    • 3
  • M. Yamato
    • 1
  1. 1.Department of Radiology, Dokkyo University School of Medicine, 880 Kitakobayashi, Mibu, Shimotsuga-gun, Tochigi-ken 321-02, Japan Tel.: 81–282–87-2171, Fax: 81–282–86–4940JP
  2. 2.Department of Paediatrics, Nihon University School of Medicine, Tokyo, JapanJP
  3. 3.Department of Paediatrics, Dokkyo University School of Medicine, Tochigi, JapanJP