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Pustulotic arthroosteitis is a relatively common disease in Japan, but coexistence of rheumatoid arthritis (RA) and pustulosis palmoplantaris (PPP) is uncommon. The case report of a 38-year-old woman illustrates a rare and poorly understood relationship between PPP and RA. The two diseases have in common a tendency of leukocyte migration, which is the basis of a hypothesis of a related cause.
Motilities of leukocytes in response to bacterial antigens or sera were examined in tissues from patients with palmoplantar pustulosis (PPP). Accelerated migration towards bacterial antigens was detected in the case of Staphylococcus epidermidis in 9 of 19 patients, to Propionibacterium acnes in 5 of 19, to Proteus mirabilis and Staphylococcus aureus in 3 of 19, while no acceleration was found in neutrophils from the controls. A significantly accelerated migration of normal lymphocytes in response to both patients' and control sera was nil. Inhibition of migration of guinea pig peritoneal exudate cells mixed with lymphocytes from PPP patients was detected in 12 out of 14 patients with the addition of S. epidermidis antigen and 8 of 14 with the addition of P. acnes antigen, while no such inhibition was detected in all 7 controls. The accelerated migration of neutrophils and inhibition of macrophages may participate in the development of PPP.
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Compositae dermatitis occurred in a 9-year-old boy with a strong personal and family history of atopy. Positive patch test reactions were 2+ for dandelion (Taraxacum officinale), false ragweed (Ambrosia acanthicarpa), giant ragweed (Ambrosia trifida), short ragweed (Ambrosia artemisifolia), sagebrush (Artemisia tridentata), wild feverfew (Parthenium hysterophorus), yarrow (Achillea millifolium), and tansy (Tanacetum vulgare) and 1+ for Dahlia species and English ivy (Hedera helix). Patch tests were negative for another 30 plants, including cocklebur (Xanthium strumarium), dog fennel (Anthemis cotula, fleabane (Erigeron strigosus), sneezeweed (Helenium autumnale), and feverfew (Tanacetum parthenium). The eruption resembled atopic dermatitis morphologically but was prominent on the palms and face and dramatically spared the area of the boy's feet covered by his shoes. The condition has always been seasonal, worsening in summer, especially July, and it clears on avoidance of contact. This case is believed to represent a contact dermatitis to oleoresins of Compositae plants; inhalants as a cause of systemic aggravation are not likely to be important in this patient.
We report two patients with infantile acropustulosis; one patient with a history of atopic dermatitis and the second with an abnormally high IgE serum value. Atopy has not been reported previously in patients with infantile acropustulosis. Immunofluorescent studies of involved skin gave negative results.
Two cases of inflammatory joint disease during attacks of aseptic palmo-plantar pustulosis are reported. Both patients had vertebral involvement: disco-vertebral erosions at several levels in one case and spondylitis and intersomatic ossification in the other. One patient also had sacroiliitis and involvement of the anterior thoracic skeleton (costoclavicular joints, costal cartilages ans xiphoid) confirmed by bone scintigraphy. The relation between this cutaneous-articular syndrome and other inflammatory joint diseases, especially ankylosing spondylitis and psoriatic arthropathy are discussed. The very unusual involvement of the anterior thoracic skeleton, the coexistence of aseptic palmo-plantar pustulosis and absence of antigens HLA B27, B13 and B17 suggest a similarity to the pustular osteoarthritis recently described by Japanese workers who grouped this condition with other spondylarthropathies.
The cases of nineteen patients with sternocostoclavicular hyperostosis were reviewed retrospectively. Of particular interest were the responses to antibiotics and prostaglandin inhibitors. The prostaglandin inhibitors relieved the pain within three to four weeks in sixteen of eighteen patients so treated. However, the inhibitors gradually became less effective in most patients. Oral antibiotics were more effective than the inhibitors in relieving the pain of eight of the eleven patients who were given antibiotics. Pustulosis palmaris and plantaris, commonly associated with sternocostoclavicular hyperostosis, diminished after antibiotic therapy, as did the chest pain in most patients. The similarities between the age and sex distributions and the responses to antibiotics of the patients with sternocostoclavicular hyperostosis and those with pustulosis suggest that these disorders have a common etiology, and that the pustulosis may be a so-called bacterid reaction and the hyperostosis, a manifestation of a systemic reaction to a focal infection.
Unusual, discrete palmar and plantar symptoms observed in thirty of forty-seven children with dermatitis herpetiformis are described. The diagnosis was verified in every case by the demonstration of granular IgA deposits in the skin. Forty-five of the children showed villous atrophy in jejunal biopsy specimens. In four cases extensive, exudative, bullous palmar symptoms, similar mild plantar changes, and healing with desquamation were observed. At least once during treatment we found very discrete, reddish-brown spots or small blisters on the flexor surface of the fingers and on the palms in thirty patients. Similar lesions occurred on the soles and plantar surface of the toes in only three patients. In asymptomatic patients and those treated with either a gluten-free diet or sulfone/sulfapyridine, the phenomenon was not manifest.
A case of naevus comedonicus on the palms and soles and scalp is reported. A similar case was previously reported by Leppard. The rare localization of this folicular hamartoma is commented.
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We describe the case of a white male with chronic inflammation of one knee, the sternoclavicular area and the dorsal spine, who also had pustulosis palmaris et plantaris. Radiological changes were suggestive of pustulotic arthroosteitis. Our patient had prominent peripheral joint changes not previously emphasized.
The authors report three cases of palmo-plantar pustulosis associated with articular signs: erosive arthritis of the right first sternocostal joint in 2 cases (without hypertrophy of the clavicle or the sternum) and atlanto-occipital arthropathy with marked neck stiffness in another case. The HLA phenotype of one case was: A2 - A9 - B14 - X - DR3 - DR4. A surgical sterno-costal biopsy revealed non-specific inflammatory lesions in 2 cases. In one of these cases, a Corynebacterium sp. was isolated. The clinical course was favourable in response to local antibiotic therapy in one case (follow-up of 8 years) and after treatment with non-steroidal anti-inflammatory agents in 2 cases (follow-up of one to two years). The skin biopsy revealed non-spongiform (and therefore non-psoriatic) unilocular pustulosis, distinguishing this non-bacterial pustulosis from pustular palmo-plantar psoriasis with which it is frequently confused. These cases are similar to the cases of "pustulotic arthro-osteitis" reported by Japanese authors (Sonozaki et al.), which appear to be rare in Europe. They seem to be an early form in a vast range of spondylo-arthropathies including rheumatism and acne conglobata. The aetio-pathogenesis of this syndrome is discussed; one of the cases is strongly suggestive of an infectious origin (Corynebacterium). These lesions do not appear to be a form of reactive arthritis, as the presence of HLA B27 is rare in both the European and the Japanese cases.
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Report of a 30 years old man story pustulosis palmaris et plantaris, spondylodiscitis L5-S1, right sterno-clavicular arthritis, right sacro-iliitis, and pubic symphysitis. Multiple bacteriologic investigations did not discover any infectious articular process. Histologic articular findings were inflammatory pattern, non steroid anti-inflammatory drugs corrected the arthritis. Anterior thoracic arthropathies are quasi-constant. Inflammatory spondylodiscitis are frequent, while sacro-iliac involvement is infrequent, this pattern distinguishing pustular arthro-osteitis from classic pelvispondylitis. Radiologic lesions are peculiar by predominance on one articular side at each site. Nosology is discussed. Slow infection by non virulent germs has been suspected , but seems improbable. This affection seems close seronegative spondylo-arthropathies and reactive arthritis, like acne conglobata arthritis for instance. Usual absence of HLA-B27 antigen must be noted.
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