A complicated case of pachydermoperiostosis with spondyloarthritides: a case report

Journal of Medical Case Reports
Qing ZhangKeyi Yu

Abstract

Pachydermoperiostosis is a rare, hereditary disease commonly presenting with digital clubbing, pachydermia and periosteal hypertrophy. Therapeutic options for pachydermoperiostosis are few because of the unknown pathogenesis. Here, we report the complicated case of a patient with pachydermoperiostosis combined with spondyloarthritides, who was refractory to steroids and tumor necrosis factor alpha antagonists. We treated this patient with zoledronic acid and performed an arthroscopic synovectomy, with a satisfactory outcome. To the best of our knowledge, this is the first report in English on the combination of zoledronic acid administration and synovectomy for the treatment of a patient with pachydermoperiostosis and spondyloarthritides. An 18-year-old Han Chinese man was diagnosed with pachydermoperiostosis in the presence of digital clubbing, periostitis and swollen limbs. Combined spondyloarthritides was also considered based on his lower back pain, lower limbs synovitis, bilateral sacroiliac sclerosis and a positive test for human leukocyte antigen B27, as well as immunoglobulin A nephropathy. He was refractory to steroids and tumor necrosis factor alpha antagonists, but treated with intravenous zoledronic acid and an arth...Continue Reading

References

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Citations

Oct 30, 2016·Ophthalmic Plastic and Reconstructive Surgery·Marlos R L SilvaAntonio Augusto V Cruz
Apr 23, 2019·Oxford Medical Case Reports·Binit VaidyaShweta Nakarmi

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Methods Mentioned

BETA
biopsy
ESR

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