A case of recurrent Caffey's disease treated with naproxen. Clin Orthop Relat Res 1996 Feb(323):304-9
Date
02/01/1996Pubmed ID
8625597DOI
10.1097/00003086-199602000-00043Scopus ID
2-s2.0-0030022123 (requires institutional sign-in at Scopus site) 26 CitationsAbstract
Caffey's disease (infantile cortical hyperostosis) is considered to be a benign self-limiting disease of uncertain etiology that typically appears in early infancy. The following case of Caffey's disease in a young girl is significant from 2 standpoints. First, the course of the disease is atypical because of its severe recurrent nature. The patient experienced 6 highly symptomatic episodes of cortical hyperostosis involving multiple new sites and previously healed lesions. These recurrent episodes persisted into her fourth year of life. In general, the reported cases of late recurrence of Caffey's disease describe patients with minimal symptoms and mild bony involvement. The second area of significance involves the successful use of naproxen to control the symptoms and arrest the progression of the patient's disease. Reports of hyperostosis in patients receiving prostaglandin E to maintain a patent ductus arteriosus suggest that prostaglandins may play a role in the etiology of Caffey's disease. Given this evidence, it seems plausible that there may be a therapeutic role for prostaglandin inhibitors in the successful treatment of Caffey's disease. The patient in this case had immediate and complete resolution of her symptoms while receiving naproxen with no recurrent bone formation while being treated with antiinflammatory drugs.
Author List
Thometz JG, DiRaimondo CAAuthor
John G. Thometz MD Professor in the Orthopaedic Surgery department at Medical College of WisconsinMESH terms used to index this publication - Major topics in bold
Anti-Inflammatory Agents, Non-SteroidalCyclooxygenase Inhibitors
Female
Humans
Hyperostosis, Cortical, Congenital
Infant
Naproxen
Radiography
Recurrence
