J Rheum Dis.  2015 Apr;22(2):127-131. 10.4078/jrd.2015.22.2.127.

A Case of Palmoplantar Pustulosis Present in the Daughter of a SAPHO Syndrome Patient

• Choi JY
• Kim JM
• Song R
• Lee YA
• Lee SH
• Yang HI
• Hong SJ

• KMID: 2222917
• DOI: http://doi.org/10.4078/jrd.2015.22.2.127

Abstract

SAPHO syndrome is a rare inflammatory, pseudoinfectious disease. Initially it was an acronym for Syndrome Acne Pustulosis Hyperostosis Osteitis, and the meaning of S was later changed to synovitis. It occurs predominantly in children and adults and is not common over 60 years. The most common clinical presentation is osteoarticular involvement at the anterior chest wall and skin manifestations may be evident, but it could occur years earlier or develop later. We report on two cases of mother and daughter. A 51-year-old female was diagnosed with SAPHO syndrome with costochondritis and palmoplantar pustulosis. Five years later, her 31-year-old daughter presented with similar skin manifestations of the hand and foot.

Keyword

SAPHO syndrome; Acquired hyperostosis syndrome; Palmoplantaris pustulosis

MeSH Terms

Acne Vulgaris
Acquired Hyperostosis Syndrome*
Adult
Child
Female
Foot
Hand
Humans
Hyperostosis
Middle Aged
Mothers
Nuclear Family*
Osteitis
Psoriasis
Skin Manifestations
Synovitis
Thoracic Wall

Figure

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  Figure 1. Palmoplantar pustulosis at both hands of a 51-year-old female patient.

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  Figure 2. Increased uptake at costochondral junction at whole body bone scan (A; arrow), irregular margin and contour bulging at left 1st costochondral junction at chest computed tomography (B; arrow). L: left, R: right, Ant: anterior, Post: posterior.

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  Figure 3. Palmoplantar pustulosis at both hands of a 31-year-old female patient.

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  Figure 4. Increased uptake at both ulnocarpal joint at a 31-year-old female patient (A), normal wrist x-ray (B).

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  Figure 5. Microscopic findings of pustulosis. Hyperkeratosis (white arrow) and pustules of epidermis (black arrows) at section from skin (H&E, ×10) (A) and perivascular lymphocytic infiltration of dermis (H&E, ×20) (B).

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