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A female patient with acrodermatitis chronica atrophicans and widespread sclerodermiform skin lesions with a high IgG antibody titer against Borrelia burgdorferi is presented. The rapid improvement after high-dose penicillin G therapy and the course of the Borrelia antibody titer suggest a persistence of the causative organism.
Seven cases of acrodermatitis chronica atrophicans (ACA), 11 cases erythema chronicum migrans (EMC) and 3 cases of lymphadenosis benigna cutis (LABC) have been analysed. In Warthin-Starry stained sections, spirochetal structures were present in all cases. The spirochetes were mesodermotropic and irregularly distributed.
Using indirect immunofluorescence, IgG antibodies against the recently detected Ixodes-ricinus-spirochaeta, which causes erythema chronicum migrans could be demonstrated in all 21 persons with acrodermatitis chronica atrophicans. Titers were from 1 : 64 to 1 : 1024, specific IgM antibodies were demonstrable in only 5 patients in a titer of 1 : 64. Even after treatment with penicillin high IgG antibody titers of up to 1 : 1024 were found. Fourfold decreases could be found only once for each IgG and IgM antibodies.l Serology indicates that acrodermatitis chronica atrophicans as well as erythema chronicum migrans is caused by the Ixodes-ricinus-spirocheta.
To study the serological response to Borrelia burgdorferi after treatment of late Lyme borreliosis, consecutive serum samples from 20 patients with Borrelia arthritis and 21 with acrodermatitis chronica atrophicans were analysed with capture IgM ELISA and indirect IgG ELISA, both using B. burgdorferi flagella as antigen. Seven patients had positive IgM OD values, whereas all 41 had positive IgG OD values before therapy. In the majority, highly elevated IgG OD values were seen. All patients improved after antibiotic therapy, 32 recovering completely, while 9 had sequelae. At follow-up after 6 months to 5 years, 4/7 patients became negative IgM ELISA, whereas 3 still had slightly elevated IgM OD values 6 months, 1 year and 4.5 years, respectively, after therapy. Only one patient became negative in IgG ELISA during follow-up, although a significant decline in IgG OD values was seen in 22 of the remaining 40 initially IgG-positive patients. The serological response after successful treatment of Borrelia arthritis and acrodermatitis chronica atrophicans may persist for several years even with highly elevated IgG OD values in patients who have recovered completely.
Forty-seven patients with the late borrelial manifestation acrodermatitis chronica atrophicans (ACA) and with objective neurological and/or neurophysiological findings were followed up after antibiotic treatment with dermatological, serological, neurological and neurophysiological controls. Despite a good therapeutic effect on ACA lesions, specific antibody values and symptoms of irritative nerve lesions, the objective neurological and neurophysiological findings of nerve deficit remained unchanged. There was no progress of neuropathy findings during the follow-up time. Our interpretation of the results is that the remaining neuropathy signs after treatment of ACA are neurological sequelae and not manifestations of persisting Borrelia infection.
BACKGROUND AND DESIGN: Specific humoral and cell-mediated immune responses play an important role in the pathogenesis of Lyme borreliosis. Several previous studies demonstrated that a specific cellular immune response to Borrelia burgdorferi can occur independently of a diagnostic humoral response. Little is known about T-cell reactivities against B burgdorferi in early and late cutaneous manifestations of Lyme borreliosis. We studied the lymphoproliferative response of peripheral blood mononuclear cells to B burgdorferi antigen from 99 patients (25 with erythema migrans, 16 with acrodermatitis chronica atrophicans, 13 with lymphadenosis benigna cutis, and 45 with localized scleroderma) and 21 control subjects. The results are expressed as a stimulation index (SI) (mean count per minute of triplicate cultures with stimulant divided by mean count per minute without stimulant). The serum samples from all patients and control subjects were tested for antibodies to B burgdorferi by indirect immunofluorescence assay. RESULTS: The 21 healthy seronegative controls had an SI of 3.3 +/- 2.0 (mean +/- SD). Compared with that of control subjects, the SIs were significantly elevated in patients with erythema migrans (9.8 +/- 9.1), acrodermatitis chronica atrophicans (11.8 +/- 8.2), and lymphadenosis benigna cutis (7.2 +/- 6.2). The 45 patients with localized scleroderma had elevated proliferative responses, with an SI of 6.5 +/- 7.3, but these responses did not significantly differ from those of controls. Elevated titers of antibodies to B burgdorferi were present in six (24%) of 25 patients with erythema migrans, five (38%) of 13 patients with lymphadenosis benigna cutis, and 13 (29%) of 45 patients with localized scleroderma. All 16 patients with acrodermatitis chronica atrophicans had markedly elevated antibody titers. CONCLUSIONS: Our findings show that a significant lymphoproliferative response to B burgdorferi occurs in the majority of patients with cutaneous manifestations of Lyme borreliosis. The lymphocyte proliferation assay may be of diagnostic value in patients in whom Lyme borreliosis is strongly clinically suspected and who have nondiagnostic levels of antibodies against B burgdorferi.
Spirochetes were recovered from the skin lesions of two patients with acrodermatitis chronica atrophicans. Both patients assume that they had a tick bite. Sera from both patients showed increased IgG-antibody titers to a Borrelia burgdorferi strain isolated from the skin.
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Spirochetes were recovered from the skin lesion of 1 out of 10 acrodermatitis chronica atrophicans patients (ACA). Spirochetes from this skin isolate and from Ixodes (I.) ricinus and I. dammini spirochetes were used as antigens in indirect immunofluorescence tests. All sera from 17 ACA patients showed high antibody titers to the three antigens. Seven of the 17 sera which had the highest titers had crossreactive antibodies to treponemal antigen detectable in the FTA-ABS test. The results indicate that spirochetes are of importance for ACA and probably the causative agent of this disease. The connection between ACA and tick bites and the relationship to erythema chronicum migrans Afzelius (ECMA) and Lyme disease are discussed. The results are consistent with the hypothesis that ECMA and ACA are different manifestations of the same spirochete, with ACA as a late manifestation.
A study was made of 50 consecutive patients with untreated acrodermatitis chronica atrophicans (ACA). In all patients elevated anti-spirochetal antibody titers were found at indirect immunofluorescence and enzyme-linked immunosorbent assays, and histologically biopsies exhibited a dermal lymphocytic infiltrate with a moderate to rich admixture of plasma cells and telangiectases. Nine patients had a history consistent with spontaneously healing erythema chronicum migrans Afzelius (ECMA) on the extremity on which, after 0.5-8 years, ACA lesions developed. Eight patients had a history indicating previous cranial nerve involvement and nine had had periods of severe pains in the cervical or lumbar region. Two patients had developed ECMA, facial palsy and ACA in chronological order. In 15 patients radiographic abnormalities of joints and/or bone tissue were found. Besides ACA lesions, lichen sclerosus et atrophicus- or scleroderma-like lesions were found in six patients. The inflammatory ACA lesions were sometimes discrete and had been overlooked. Joint deformities, sclerotic lesions, diffuse edema or pain were the cardinal symptoms in some patients. The findings indicate that clinical recognition of ACA may be difficult and that a combination of clinical, histopathologic and serologic findings may be necessary to establish the diagnosis. The results are consistent with the concept that ACA is a late manifestation of infection by the same spirochete as causes ECMA and Bannwarth's syndrome.
We report on an 81-year-old woman suffering from extensive acrodermatitis chronica atrophicans with facial involvement. This unusual manifestation may be related to an immunodeficiency state in the course of multiple malignancies. The cutaneous lesions cleared significantly during an aminopenicillin therapy administered orally over 4 weeks.
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Borrelia burgdorferi is a major cause of morbidity in wooded area in western Europe and the eastern seaboard of the U.S.A. Diagnosis of late stage infection and associated disorders may be difficult and often requires an array of different diagnostic procedures. Here we report an 11-year-old girl with acrodermatitis chronica atrophicans affecting all four limbs and parts of the trunk. The diagnosis was made on the basis of clinical appearance, serological and histopathological findings, and the lesional detection of B. burgdorferi-specific gene segments by polymerase chain reaction. This very unusual, severe case illustrates that despite being a late manifestation of tick-borne B.burgdorferi infection, usually occurring in adults, acrodermatitis chronica atrophicans may already appear at a young age and may be characterized by extensive skin involvement.
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