ARTICLE
Auteur(s) : Müzeyyen Gönül1,
Arzu KIlIç1, Seray Külcü Çakmak1, Ülker
Gül1, Oğuzhan Koçak1, Murat
Demiriz2
1Ankara Numune Education and Research Hospital, 2nd
Dermatology Clinic, Ankara, Turkey
2GATA School Of Medicine, Pathology Department, Ankara,
Turkey
accepté le 21 Août 2006
Tuberculosis, one of the oldest diseases that is known, continues
to be a significant problem in developing countries [1]. Cutaneous
tuberculosis makes up a small proportion (10%) of all types of
tuberculosis and shows considerable morphological variability
according to the host immunity and the way the organism got through
the skin [1, 2]. Six different varieties of cutaneous tuberculosis
have been described: Primary inoculation tuberculosis, tuberculosis
verrucosa cutis, lupus vulgaris (LV), scrofuloderma, orificial
tuberculosis and miliary tuberculosis [3]. Lupus vulgaris is the
most common form and frequently involves the head and neck (nose or
the lobes of the ears) [1, 2, 4, 5]. We report a case of
long-standing and uncommon annular form of LV with an unusual
localization.
Case report
A 46-year-old woman attended to our clinic with progressive,
asymptomatic, annular skin lesions on her right forearm and hand
dorsum for five years. She had received many different therapies
such as antifungal, antibiotic and corticosteroid therapies for
these lesions previously. These medications were not effective and
the lesions deteriorated. On dermatological examination,
well-demarcated, irregular bordered, violaceous colored, elevated
and crusted annular lesions on her right hand dorsum and forearm
were observed ( (figure
1) ). The patient did not reveal a history of tuberculosis
in herself or any of her relatives, but before the appearence of
lesions the patient had a history of milking a cow which might be
the cause of cutaneous tuberculosis. She had a history of BCG
vaccination in her infancy. Routine laboratory tests and pulmonary
X-ray were normal. PPD revealed 20 mm induration after 48
hours. Direct microscopic examination and cultures of the tissue
samples from lesions for bacteria, fungi and Mycobacterium
tuberculosis were all negative. Tissue, sputum and urine specimens
were tested using polymerase chain reaction (PCR). A standard
protocol for extraction and amplification of Mycobacterium
tuberculosis complex DNA was performed. The primary sets used to
amplify the 123-bp IS6110 gene fragment consisted of TBC1 and TBC2
[6]. Mycobacterium tuberculosis complex was not detected in the
specimens. Although the PCR examination of the skin tissue was
performed in two different centers, negative results were
confirmed. No tuberculotic focus was detected in detailed
examination of pulmonary, genitourinary and skeletal systems and
lymph nodes. Histopathological examination revealed dermal
lymphocytic inflammatory infiltrate containing many granulomas
consistent with LV ( (figure 2) ). Periodic acid
schiff (PAS) stain was negative for deep fungal infection and
Giemsa stain was negative for cutaneous leishmaniasis. Although the
tuberculosis culture and PCR did not confirm the diagnosis we
decided to administer antituberculous therapy according to the
clinic and histopathologic findings of the lesions. The therapy
consisted of isoniazid 300 mg/d, rifampicin 600 mg/d, ethambutol
25 mg/kg/d and pyrazinamide 1 500 mg/d in the first two
months. Later, the therapy was continued with isoniazid and
rifampicin (same dosages) for seven months. Regression of the
lesions was observed in the second week of therapy.
Discussion
The most common type of cutaneous tuberculosis is LV. More than 80%
of the lesions of LV are on the head and neck, particularly around
the nose and ear lobes [1, 2, 4, 5, 7]. Localization on the forearm
as in our case is unusual. The lesions start as a tiny, red-brown,
soft, flat papules, and progress to larger plaques by peripheral
enlargement. The disease is marked by ulceration and scarring [5,
7]. The annular form of LV lesions as in our case is rare.
Werschler et al reported a case of annular LV which was localized
on the face [8]. In our case the annular lesions were localized on
the arm, which is an uncommon area for LV. The diagnosis of annular
form is difficult and must be differentiated from the other annular
diseases such as tinea infection, psoriasis, annular elastolytic
giant cell granuloma and annular erythemas [2]. Our case was
treated previously with antifungals, antibiotics and
corticosteroids for a long time but the lesions deteriorated. The
differential diagnosis of the lesions from tinea infection,
psoriasis and annular erythemas was confirmed by negative KOH
examination and the existence of granulomas in the biopsy specimen.
We excluded the diagnosis of annular elastolytic giant cell
granuloma due to the loss of multinucleated giant cells containing
remnants of elastic fibres in conjunction with a loss of elastic
fibres in dermis histopathologically.
Histopathological examination, mycobacterial culture, PPD test,
and PCR (The sensitivity is around 50%) are important methods for
the diagnosis of cutaneous tuberculosis [7, 9-12]. Microbial
culture in LV is frequently negative, only 6% positivity of
cutaneous cultures from patients with LV has been reported.
Histopathological studies show tubercules or tuberculoid granulomas
with slight or absent caseation in the papillary dermis and a
variable degree of epidermal hyperplasia [7]. In our case,
mycobacterial culture and PCR were negative. PPD was positive
(20 mm). Histopathological examination revealed dermal
lymphocytic inflammatory infiltrate containing many granulomas.
Depending on clinical and histopathological findings, LV was
diagnosed and the success of the treatment confirmed the diagnosis.
Negative PAS staining for deep fungal infection, negative Giemsa
staining for cutaneous leishmaniasis and negative PCR for other
atypical mycobacterial infections supported diagnosis of LV. The
lesions significantly regressed with antituberculous therapy
without scarring.
LV usually results from a reinfection of the skin in people with
a high degree of tuberculin sensitivity and mostly occurs with
hematogeneous, lymphatic or direct spread from visceral
tuberculosis. In rare instances it can appear at the site of
primary inoculation such as the site of BCG vaccination [7]. Due to
the history of cow milking, hand and forearm localization of the
lesions and lack of any other focal tuberculotic focus despite
detailed examination, we thought that the patient was infected by
primary inoculation.
Tuberculosis is still a serious problem in both developing and
developed countries. It is often confused with various cutaneous
disorders both clinically and histopathologically, due to
considerable morphological variability [1, 2]. Mycobacterial
infection is a common complication in immunosuppressed patients and
in these cases, LV can present with an uncharacteristic manner.
However as our case was not immunosuppressed, it is interesting
that LV presented in an annular form with an uncommon
localization.
We report a case of long-standing, undiagnosed and uncommon form
of lupus vulgaris with an unusual localization. We think that the
diagnosis of LV should be kept in mind in cases with long standing
skin lesions which do not respond to the routine treatments, even
if the results of tuberculosis culture and PCR are negative.
Acknowledgements
Financial support: None. Conflict of interest: None.
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