Ting Xiao1, Hong-Guang Lu2, Jian Wu1, Chun-Di He1 and Hong-Duo Chen1*

Department of Dermatology, 1No. 1 Hospital of China Medical University, 155 North Nanjing Street, Shenyang 110001 and 2Affiliated Hospital of Guiyang Medical Collage, Guiyang, China. *E-mail: chenhd@cae.cn.

The first two authors have contributed equally to this paper and should be considered as first author.

Accepted August 28, 2008.

Sir,

Pustular bacterid (PB) is a pustular disorder of the hands and/or feet. It was first described by Andrews and co-workers (1, 2). There is controversy as to whether PB is a separate disease or is a variant of psoriasis pustulosa palmaris et plantaris (PPP). We describe here a case of PB in a patient with ankylosing spondylitis (AS). This is the first report of PB associated with AS and supports the theory that PB is a distinct disease.

CASE REPORT

A 42-year-old Chinese woman presented on 16 August 2005 with a one-week history of pustules on both hands, a fever of approximately 38°C, sore throat, cough and expectoration with yellow-coloured sputum. She had a 13-year history of painful orthopaedic symptoms with morning stiffness in the sacro-iliac area, which improved with exercise. AS had been diagnosed according to characteristic clinical, X-ray and computed tomography (CT) findings in the Department of Rheumatology of the same hospital approximately one month previously. She had been prescribed oral sulfasalazine 250 mg t.i.d., leflunomide 10 mg q.d. and celecoxib 200 mg b.i.d. Her pain had relieved significantly within one week and she was discharged after 11 days, continuing the 3 medications until admitted to our department. She had no personal or family history of psoriasis.

Physical examination revealed multiple isolated 2–8 mm diameter pustules on both hands, especially the palms (Fig. 1). The feet and other parts of body were spared. No fissured or geographic tongue was observed.

Fig. 1. (A, B) Multiple isolated 2–8 mm diameter pustules on both hands, especially the palms.

Laboratory tests, including blood, urine and stool routine tests, liver and kidney function tests, fast blood glucose and electrolytes were normal. Serum IgG, IgA, IgM and complement levels were normal. Serum anti-streptolysin antibodies, C-reactive protein, rheumatoid factors, antinuclear antibodies, anti-dsDNA antibodies, anti-extractable nuclear antigen antibodies, anti-cardiolipin antibodies and anti-neutrophil cytoplasmic antibodies were negative. HLA-B27 were negative. The erythrocyte sedimentation rate was 21 mm/h. Serum antibodies against Mycobacterium tuberculosis, Mycoplama pulmonis, human immunodeficiency virus, herpes simplex virus, rubella virus, toxoplasma virus, cytomegalovirus, respiratory syncytial virus, Epstein-Barr virus, coxsackie virus, adenovirus and hepatitis A, B, C and E viruses were negative. Serum hepatitis B surface antigen was negative. Repeated cultures from contents of pustules, sputum and blood for bacteria and fungi were negative. Throat smears revealed many Gram-positive cocci. Throat swab cultures revealed growth of group A β-haemolytic Streptococcus.

PB and acute upper respiratory tract infection were clinically diagnosed. Intravenous azithromycin was administered as a single daily dose of 10 mg/kg for 3 days. The pustules resolved dramatically overnight (Fig. 2). The fever, sore throat, cough and expectoration were also relieved. The patient refused a biopsy and requested a discharge. The pustules crusted over completely after 3 days’ azithromycin therapy and the patient was then discharged. No relapse of pustules occurred during a 2-year follow-up.

Fig. 2. (A, B) Most of the pustules crusted after a single dose of 500 mg azithromycin.

DISCUSSION

To date, the aetiology of PB is not clear. Many authors have found that PB is triggered by upper respiratory tract infections (3). PB has been reported to be associated with sternocostoclavicular hyperostosis or Tietze’s syndrome (4, 5). Occasionally, PB can be generalized (6). In the present case, it seems that the PB might be related to AS, as the patient had a long history of AS, which could affect her immune status, resulting in susceptibility to infections.

Many authors still question whether PB is a distinct disease. It is difficult to differentiate PB histopathologically from psoriasis PPP. Bacharach-Buhles suggested that “true” PB had the following features: coincidence with an infection, no personal or family history of psoriasis, isolated sterile pustules, no lesions of psoriasis vulgaris, good response to antibiotics and a short course (3). The present case fulfilled the above features of PB and had clinical manifestations fairly distinct from those of psoriasis PPP. This case provides further evidence that PB might be a distinct entity.

REFERENCES

1. Andrews GC, Machacek GF. Pustular bacterids of the hands and feet. Arch Dermatol Syphiol 1935; 32: 837–847.

2. Andrews GG, Barnes MC. Pustular bacterids and allied conditions. South Med J 1941; 34: 1260–1265.

3. Bacharach-Buhles M, el Gammal S, Altmeyer P. The pustular bacterid (Andrews). Are there clinical criteria for differentiating from psoriasis pustulosa palmaris et plantaris? Hautarzt 1993; 44: 221–224.

4. Chigira M, Maehara S, Nagase M, Ogimi T, Udagawa E. Sternocostoclavicular hyperostosis. A report of nineteen cases, with special reference to etiology and treatment. J Bone Joint Surg Am 1986; 68: 103–112.

5. Ishibashi A, Nishiyama Y, Endo M, Kawaji W, Kato T. Orthopedic symptoms in pustular bacterid (pustulosis palmaris et plantaris): Tietze’s syndrome and arthritis of manubriosternal joint due to focal infection. J Dermatol 1977; 4: 53–59.

6. Miyachi Y, Danno K, Yanase K, Imamura S. Acute generalized pustular bacterid and immune complexes. Acta Derm Venereol 1980; 60: 66–69.