Rouanet, Isabelle; Jantac, Marie; Lechiche, Catherine; Hope-Rapp, Emilie; Sotto, Albert
Department of Infectious Diseases and Tropical Medicine, University Hospital of Nîmes, Place du Professeur Robert Debré, Nîmes 30029 Cedex 9, France.
Correspondence to Albert Sotto, Department of Infectious Diseases and Tropical Medicine, University Hospital of Nîmes, Place du Professeur Robert Debré, Nîmes 30029 Cedex 9, France. Tel: +33 4 66 68 41 49; fax: +33 4 66 68 40 21; e-mail: email@example.com
Received 21 January, 2012
Accepted 30 January, 2012
Neutrophilic eccrine hidradenitis (NEH) is a neutrophilic dermatosis reported for the first time in 1982 by Harrist et al.[1,2]. About 50 cases have been reported in the literature to date. Most of the time, NEH is associated with underlying hematologic or neoplasic disease. We report a case of NEH in an HIV-infected patient without other underlying disease.
A 63-year-old man, HIV positive since 1995, with CD4 lymphocytes count of about 600/mm3, undetectable viral load and highly-active antiretroviral therapy (raltegravir, atazanavir and ritonavir), was hospitalized in May 2011. He developed a fever (38–39.5°C) for 3 weeks, associated with shivers, myalgia, lost of weight (3 kg) and diarrhea. A few days later, a cutaneous eruption appeared consisting of erythematous (red or violaceous), nonpruriginous, painless, infiltrated papules, beginning on the trunk, then extending to the upper limbs. Physical examination was otherwise unremarkable.
Biological tests noted anemia (10.3 g/dl), thrombocytemia (523000/mm3), and hyperleukocytosis (41900 white blood cells/mm3 with 90% of neutrophils). C-reactive protein, β-2 microglobulin, haptoglobin, ferritinemia, and fibrinogen were increased. Renal function, aspartate amino transferase and alanine amino transferase were normal; γ-glutamyl transpeptidase and alkaline phosphatase were three times normal. CD4 lymphocyte count was 376/mm3 (33%) and HIV viral load was undetectable. Infectious checkup was negative. Chest radiograph, abdominal echographia, PET/computed tomography and osteomedullar biopsy were normal. Histological examination of cutaneous lesions showed, in the lower dermis, a polymorphous and moderate inflammatory infiltrate surrounding and infiltrating eccrine coils and ducts with lymphohistiocytes and numerous neutrophils. Neither leucocytoclasia nor epithelioid granulomia were seen. Histological examination confirmed the NEH diagnosis.
Treatment with colchicine (1 mg per day) was administered, resulting in apyrexia and rapid healing of the lesions and other symptoms. Two weeks later, the patient was quite asymtomatic, and only some myalgia persisted. Biological parameters were improving too. The patient then stopped the treatment by himself after 4 weeks. Seven months later, the patient was asymptomatic and biological tests were normal.
The clinical and histological data in the present case are quite characteristic of NEH . Typical cutaneous lesions are well delimited, infiltrated or edematous papules, or plaques of variable size, asymptomatic or painful, usually erythematous but sometimes pigmented or purpuric. They can be unique or multiple, develop on the upper trunk, upper limbs, face (periorbital areas), or in a distal disposition. Fever is frequently observed. Spontaneous evolution is healing without after effects in a few days.
In 90% of cases, NEH develops in patients with underlying malignancies. It often appears within 10 days after the beginning of a chemotherapy treatment, so NEH is considered in relation with the treatment. Sometimes, NEH heralds the onset of malignancy or relapse, and is considered as paraneoplastic phenomenon. NEH can also occur after growth colony-stimulating factor (G-CSF), or during inflammatory disease (as Behcet's disease). To our knowledge, five cases of NEH in HIV-infected patients were reported [3–7]. For three of them, the role of antiretroviral treatment was mentioned: zidovudine in two cases and zalcitabine in one [3,4,6]. Another case was described during a Pneumocystis carinii pneumonia in a patient receiving numerous treatments without antiretroviral therapy . In one case, NEH heralded the diagnosis of non-Hodgkin's lymphoma . Our case was unique in that it represented a case of NEH associated with new antiretroviral agents (protease inhibitor and integrase inhibitor) and was without any underlying disease.
The pathogenesis of NEH is still poorly understood and two major hypotheses were proposed. NEH could be the consequence of a direct cytotoxicity of drugs secreted in the sweat on eccrine coils and ducts cells. Intradermal injections of bleomycin into normal skin induce histological lesions close to NEH. Other investigators postulate that NEH could be a paraneoplastic phenomenon similar to other neutrophilic dermatoses. Cases induced by colony-stimulating factors suggest that NEH could be provoked by stimulation of neutrophilic progenitor proliferation and chemotaxis by G-CSF.
There is no specific treatment for NEH, but nonsteroidal antiinflammatory drugs have been attempted, resulting in rapid healing of the lesions and apyrexia. Corticosteroids appear to shorten the duration of lesions and to relieve pain and fever in a few cases. Colchicine has been efficient in one case of idiopathic NEH . Dapsone was successfully used in one instance of recurrent NEH .
The pathophysiology of the NEH does not appear to be unique. The occurrence of NEH in HIV-infected patients raises the role either of HIV or antiretroviral treatment through a mechanism that could be close to those described with antimitotic treatments.
Conflicts of interest
There are no conflicts of interest.
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