ARTICLE
Auteur(s) : Paolo GISONDI, Marie PEREZ, Emanuela
GUBINELLI, Barbara COCUROCCIA, Marcello FAZIO, Giampiero
GIROLOMONI
Istituto Dermopatico dell‘Immacolata, IRCCS, Via Monti di Creta
104, 00167 Rome, Italy.
Reprints : G Girolomoni. Fax : (+ 39) 06‐6646‐4705
E‐mail : giroidi.it.
Article accepted on 09\06\2003
Lupus vulgaris (LV) is the most common type of cutaneous
tuberculosis in western countries [1‐4]. The typically affected
areas are the head and the neck, and less commonly, the trunk and
the scalp. The initial lesion is a reddish‐brown soft papule with a
smooth surface, that by diascopy has an apple‐jelly appearance. LV
usually consists of a single or a few papular lesions that extend
gradually to form plaques. Less common variants include
hypertrophic, ulcerative and vegetative forms [3]. In very rare
instances, LV may present with multiple lesions affecting the face,
trunk and extremities [4]. We report on two elderly patients with
disseminated LV in whom no overt immunodepression was evident.
However, in one patient dissemination occurred following an
inappropriate long term treatment with topical corticosteroids.
Case reports
Case 1. A 63‐year‐old Caucasian man presented with
asymptomatic reddish‐brown nodules and plaques localized on the
nose, trunk, right leg and foot (Fig. 1A‐1D). The lesions
had appeared firstly on the right sole 10 years before and had
been repeatedly misdiagnosed as plaque psoriasis, and treated for
many years with potent topical corticosteroids. In the last two
years, other plaques appeared on the right leg, trunk and finally
on the nose. The patient had had pulmonary tuberculosis at the age
of 30 years. He suffered from essential hypertension and
ischemic myocardial disease since the age of 57 years. On
examination, the right sole showed a large hyperkeratotic and
erythematous plaque. Moreover, multiple reddish‐brown papules and
nodules of gelatinous consistency were distributed on the back of
the right foot, the right leg, the back, as well as on the nose and
the upper lip. Histology of biopsy specimens showed typical
confluent tuberculoid granulomas in the dermis, with caseating
necrosis and numerous Langerhans‐type giant cells. No acid fast
bacilli were detected using Ziehl‐Neelsen staining, but DNA
sequences specific for Mycobacterium tuberculosis were
detected in biopsies from different lesions by polymerase chain
reaction (PCR) analysis, using a commercially available kit (Maxim
Biotech Inc., San Francisco, CA). Routine laboratory tests were
within normal ranges. Purified protein derivative (PPD) test showed
an intense reaction. Chest X‐ray did not reveal any signs of active
tuberculosis, but apical scarring fibrosis. A total body CT scan
and abdominal ultrasonography did not show abnormal findings.
Repeated urine cultures were negative for M. tuberculosis.
The patient was treated with a combination of isoniazid,
pyrazinamide and rifampicin for two months but he suffered severe
nausea induced by isoniazid, which was then replaced with
ethambutol. The plaques improved dramatically after two months and
were completely regressed after six months.
.
Case 2. A 77‐year‐old Caucasian woman presented with an
asymptomatic scarring alopecia on the occipital area and other
asymptomatic plaques on the inguinal regions. On examination, a
large reddish‐brown alopecic plaque was present on the
parieto‐occipital and latero‐cervical left regions (Fig. 2A). Moreover, similar
nodules and plaques were present on the right inguinal area and
internal and inner part of the left thigh. These last lesions had
appeared two years before our observation, whereas cervical lesions
developed during the last four months. The patient underwent
partial thyroidectomy at the age of 50 years, and on that
occasion an enlarged cervical lymph node was excised. Histologic
examination revealed a tuberculous adenitis. Histology of multiple
skin specimens showed typical tuberculoid granulomas (Fig. 2B). No acid fast
bacilli were detected by Ziehl‐Neelsen staining, but M.
tuberculosis DNA was identified by PCR analysis. Screening for
extracutaneous tuberculosis was negative. PPD test showed an
intense reaction. The patient was treated with a combination of
isoniazid, pyrazinamide and rifampicin with complete clearing of
the lesions after five months.
.
Discussion
The prevalence of tuberculosis is increasing in developed
countries due to increased immigration from countries at higher
prevalence and to the contribution of HIV coinfection [2, 5‐7].
Although a parallel increase of extrapulmonary tuberculosis has
been documented, no epidemiological data on the current prevalence
of cutaneous forms are available. LV is the commonest form of
cutaneous tuberculosis in Europe and the United States, and it may
result from hematogenous, lymphatic or contiguous spreading of a
tuberculous lesion or a clinically inapparent tuberculous focus
localized elsewhere [4, 7]. Rarely, LV follows primary inoculation
tuberculosis or Bacille Calmette‐Guerin (BCG) vaccination. LV
usually manifests with single reddish‐brown soft plaques. The face
and the neck are the most commonly affected sites in Western
countries [4, 8]. Next in frequency are the arms and legs, whereas
the involvement of the trunk and the scalp is rare. The lower
extremities, especially the buttocks, are more often involved in
the tropics and subtropics [1, 2]. We report two elderly patients
with numerous, disseminated LV lesions. Patient n. 1 had a
diffuse involvement of the right foot with a psoriasiform
appearance of the plantar lesions, which to the best of our
knowledge has been never described before. Indeed, this lesion had
been misdiagnosed as psoriasis, and the patient had applied topical
steroids for years, a time during which LV lesions diffused on the
lower limb, trunk and finally the face. Local immunodepression
induced by prolonged local steroid therapy may have favored the
local and hematogenous dissemination of the infection. Topical
glucocorticoids exert potent immunosuppressive effects by depleting
the skin of mast cells and Langherans cells, and may reduce the
capacity of resident skin cells to produce antimicrobial peptides
[9]. Patient n. 2 had LV involving the scalp and resulting in
scarring alopecia, nowadays a very uncommon presentation of LV,
which is due to the extension of the granulomatous infiltrate
deeply around the follicles [10, 11]. In both cases, Ziehl‐Neelsen
staining was negative, as frequently reported in LV [3, 4, 12], and
diagnosis, besides histology, was confirmed by PCR analysis.
Isolation of M. tuberculosis by culture is the single most
important procedure for the diagnosis of cutaneous tuberculosis,
and it is required to determine antibiotic resistance. In our
patients, mycobacterial culture was not performed because of the
positivity of PCR analysis, and because the prevalence of the
resistance to antituberculosis drugs has been found to be very low
in Italy [13]. The identification of DNA sequences of M.
tuberculosis by PCR in various clinical samples including
blood, sputum, gastric aspirate and skin is a valid tool for the
diagnosis of the disease. However, sensitivity of PCR has been
estimated to be comparable or only slightly superior to culture,
with a detection rate ranging from 53 to 77% in lesional skin
[12, 14, 15]. Therefore, clinical decision in the management of
cutaneous tuberculosis should not be based only on PCR result, but
integrated with the history, clinical and histological findings,
and culture results.
Although of rare occurrence in Western countries, cutaneous
tuberculosis remains an important diagnostic possibility in
patients with chronic cutaneous granulomatous disease. The
diagnostic challenge may be particularly relevant in the presence
of unusual clinical presentation as in the cases reported here.
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