Reumatología Clínica (English Edition) Reumatología Clínica (English Edition)
Reumatol Clin 2017;13:115-7 - Vol. 13 Num.2 DOI: 10.1016/j.reumae.2016.01.009
Images in Clinical Rheumatology
Fever, Palmoplantar Pustules and Oligoarthritis in a Young Woman
Fiebre, lesiones vesículo-ampollosas y oligoartritis en una paciente joven
Francisco Gallo Puelles, , Manuel José Moreno Ramos, María José Diaz Navarro, Manuel Castaño Sánchez
Servicio de Reumatología, Hospital Clínico Universitario Virgen de la Arrixaca, El Palmar, Murcia, Spain
Received 10 November 2015, Accepted 12 January 2016
Case Report

The patient was a 41-year-old woman, with no other remarkable findings of interest. She presented with a 3-week history of fever, arthralgia, swollen neck and pustular lesions on her hands, accompanied by pain. Over the last few days, she reported parasternal swelling that also affected right shoulder.1 Physical exploration revealed erythema and edema in neck, glenohumeral and right sternoclavicular arthritis, and diffuse dorsopalmar pustular lesions on her hands (up to 2mm) (Figs. 1–3), outer ear (Fig. 4) and neck and back (with acne). We found no other signs or lymphadenopathy.

Fig. 1.
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Diffuse pustular lesions on the dorsum of the hands.

Fig. 2.
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Diffuse pustular lesions on the dorsum of the hands.

Fig. 3.
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Diffuse pustular lesions on the palms of the hands.

Fig. 4.
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Pustular lesion on right outer ear.

Diagnosis/disease Course

Laboratory findings: leukocytosis 13,000 (normal: 10,020); platelets 68,000; C-reactive protein (CRP) 14.7 (<0.5mg/dL); and erythrocyte sedimentation rate 20mm/1st hour. Moreover, antinuclear antibodies, anti-DNA antibodies, antineutrophil cytoplasmic antibodies, anti-extractable nuclear antigen antibodies, angiotensin-converting enzyme, rheumatoid factor, anti-cyclic citrullinated peptide antibodies, immunoglobulins, complement and TSH/free T4 were normal. Mantoux and serology (hepatitis B and C viruses, human immunodeficiency virus, syphilis) and cultures were negative (blood culture, urine culture, exudates from pharynx, urethra and pustules).2–4 Neck and abdominal ultrasound and chest and shoulder radiographs provided no useful evidence. Biopsy of the skin lesions: compatible with palmoplantar pustulosis. Bone scintigraphy: increased symmetrical uptake in sternoclavicular “bull's head”, compatible with synovitis, acne, pustulosis, hyperostosis and osteitis (SAPHO) (Fig. 5). Treatment was begun with nonsteroidal anti-inflammatory drugs, systemic (16mg oral methylprednisolone) and topical (betamethasone-gentamycin ointment) corticosteroids. The fever, joint involvement and skin lesions remitted (scaling; Fig. 6), and the analytical findings were normalized (previous CRP, leukocytosis and thrombocytopenia). Once corticosteroid therapy had been reduced, methotrexate was begun with a rapid dose escalation, and the response was positive and tolerance was good.2,3,5,6

Fig. 5.
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Resolution of pustular lesions on the palms of the hands (scaling phase).

Fig. 6.
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Bone scintigraphy with typical sternoclavicular uptake of the so-called “bull's head”, compatible with synovitis, acne, pustulosis, hyperostosis and osteitis (SAPHO) syndrome.

Discussion

The SAPHO syndrome is an uncommon disorder of unknown etiology, that affects young men and women in a similar distribution.6,7 Most authors classify it under the spondyloarthritides, although it could be a reactive arthropathy secondary to an infection by an agent with a low virulence.2,4,7,8 On occasion, it may prove difficult to diagnose because it overlaps with other infectious conditions that present with fever and skin lesions.8,9 The presence of edema in neck and chest, acne and oligoarthritis, in the absence of bacteriological isolation, leads us to suspect an autoimmune etiology.10 The skin biopsy and scintigraphic data subsequently confirmed our presumed diagnosis.

Ethical DisclosuresProtection of human and animal subjects

The authors declare that the procedures followed were in accordance with the regulations of the relevant clinical research ethics committee and with those of the Code of Ethics of the World Medical Association (Declaration of Helsinki).

Confidentiality of data

The authors declare that they have followed the protocols of their work center on the publication of patient data.

Right to privacy and informed consent

The authors have obtained the written informed consent of the patients or subjects mentioned in the article. The corresponding author is in possession of this document.

Conflicts of Interest

The authors declare they have no conflicts of interest.

References
1
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Case of SAPHO syndrome with significant sternocostoclavicular hyperostosis [Article in Japanese]
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2
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SAPHO syndrome [Article in French]
Rev Med Interne, 35 (2014), pp. 595-600 http://dx.doi.org/10.1016/j.revmed.2013.12.018
3
A. Rosero,R. Ruano,M. Martín,C. Hidalgo,J. García-Talavera
Acute venous thrombosis as complication and clue to diagnose a SAPHO syndrome case. A case report
Acta Reumatol Port, 38 (2013), pp. 203-206
4
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Pustulotic arthro-osteitis associated with palmoplantar pustulosis
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Treatment of pain in SAPHO (synovitis, acne, pustulosis, hyperostosis, and osteitis) syndrome
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The SAPHO syndrome
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T.E. Carranco-Medina,C. Hidalgo-Calleja,I. Calero-Paniagua,M.D. Sánchez-González,A. Quesada-Moreno,R. Usategui-Martín
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Reumatol Clin, 11 (2015), pp. 108-111 http://dx.doi.org/10.1016/j.reuma.2014.07.003
8
S. Carneiro,P.D. Sampaio-Barros
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Rheum Dis Clin North Am, 39 (2013), pp. 401-418 http://dx.doi.org/10.1016/j.rdc.2013.02.009
9
B.K. Kundu,A.K. Naik,S. Bhargava,D. Srivastava
Diagnosing the SAPHO syndrome: a report of three cases and review of literature
Clin Rheumatol, 32 (2013), pp. 1237-1243 http://dx.doi.org/10.1007/s10067-013-2251-1
10
R.C. Zuo,D.M. Schwartz,C.C. Lee,M.J. Anadkat,E.W. Cowen,H.B. Naik
Palmoplantar pustules and osteoarticular pain in a 42-year-old woman
J Am Acad Dermatol, 72 (2015), pp. 550-553 http://dx.doi.org/10.1016/j.jaad.2014.07.014

Please cite this article as: Gallo Puelles F, Moreno Ramos MJ, Diaz Navarro MJ, Castaño Sánchez M. Fiebre, lesiones vesículo-ampollosas y oligoartritis en una paciente joven. 2017;13:115–117.


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Reumatol Clin 2017;13:115-7 - Vol. 13 Num.2 DOI: 10.1016/j.reumae.2016.01.009