Skin and bone lesions (dermato-osteolathyrism), possible side effects of D-penicillamine treatment, in a boy with cystinuria.

Abstract

A 2 1/4 year-old boy was treated for cystinuria and urolithiasis with high fluid intake, sodium bicarbonate, and D-penicillamine, over a period of 5 3/4 years, unauthorized interruptions and prescribed pauses included. Therapy was partially sucessful but regrowth of calculi coincided with interruptions of D-penicillamine administration and also with the institution of a low-dose D-penicillamine regime. Flat feet, scoliosis, pectus carinatum, hypermobility of joints, molluscoid pseudotumors and atrophic scars were alarming side effects of D-penicillamine. However, the possibility was not excluded that a forme fruste of an Ehlers-Danlos syndrome preexisted in this boy and was effected by D-penicillamine. Only the molluscoid pseudotumors regressed when D-penicillamine was reduced or omitted temporarily. Osteolathyrism caused by D-penicillamine has hitherto not been reported in man.

• Authors: B Steinmann, A Otten, R Gitzelmann
• Journal: Helvetica paediatrica acta (Helv Paediatr Acta) Vol. 34 Issue 3 Pg. 281-91 ( 1979) ISSN: 0018-022X [Print] Switzerland
• PMID: 159271 (Publication Type: Case Reports, Journal Article)

Chemical References

• Bicarbonates
• Penicillamine

Topics

• Atrophy
• Bicarbonates (therapeutic use)
• Bone Diseases (chemically induced)
• Bone and Bones (drug effects)
• Child, Preschool
• Cystinuria (drug therapy)
• Drug Eruptions
• Ehlers-Danlos Syndrome
• Flatfoot (chemically induced)
• Humans
• Joint Diseases (chemically induced)
• Kidney Calculi (drug therapy)
• Lathyrism (chemically induced)
• Male
• Penicillamine (adverse effects, therapeutic use)
• Recurrence
• Scoliosis (chemically induced)
• Skin (drug effects, pathology)
• Skin Diseases (chemically induced)
• Urinary Bladder Calculi (drug therapy)