Two cases of SAPHO syndrome accompanied by classic features of Behcet's disease and review of the literature.
We describe two patients with SAPHO (synovitis-acne-pustulosis-hyperostosis-ostitis) syndrome who presented some of the classic features of Behcet's disease. The first case is a man diagnosed as SAPHO at 74 years old. His major complaint is pain and swelling of the bilateral sterno-clavicular region for more than 14 years. Another conspicuous complication was bilateral glaucoma and episodes of iritis were recognized during the follow-up period. The second case is a 65-year-old woman, who first consulted us with right knee pain. As she had a past history of palmoplantar pustulosis and anterior chest pain, her sterile knee arthritis was diagnosed as SAPHO. She also had been suffering from recurrent oral aphthous ulceration since 6 months before visiting our hospital. Considering the clinical courses of our two cases and a review of five previously reported cases, these conditions may imply that classic features of Behcet's disease are minor complications of SAPHO syndrome. Human leukocyte antigen typing and frequent association of sacroiliitis in our cases and in the review of the literature for SAPHO syndrome with some of the classic features of Behcet's disease may indicate this condition to be a closely related disease with seronegative spondylo-arthritis.
Yabe
Hiroki
H
Department of Orthopaedic Surgery, National Hospital Organization Tokyo Medical Center, Higashigaoka 2-5-1, Meguro-ku, Tokyo, 152-8902, Japan. yabehiroki@nyc.odn.ne.jp
Takano
Yoji
Y
Nomura
Eiki
E
Nakayama
Masanori
M
Kihara
Michiya
M
Miyakawa
Shun-Ichi
S
Horiuchi
Yukio
Y
D020083:Acquired Hyperostosis Syndrome / Q000150:complications* / Q000188:drug therapy / Q000473:pathology; D000368:Aged; D000894:Anti-Inflammatory Agents, Non-Steroidal / Q000627:therapeutic use; D001528:Behcet Syndrome / Q000150:complications* / Q000188:drug therapy / Q000473:pathology; D005260:Female; D005901:Glaucoma / Q000209:etiology / Q000473:pathology; D006801:Humans; D015576:Hyperostosis / Q000209:etiology / Q000473:pathology; D007500:Iritis / Q000209:etiology / Q000473:pathology; D008297:Male; D008875:Middle Aged; D010026:Osteosclerosis / Q000209:etiology / Q000473:pathology; D016896:Treatment Outcome
D000894:Anti-Inflammatory Agents, Non-Steroidal
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