A rare case of multiple lupus vulgaris in a multifocal tuberculosis pediatric patient.

Multifocal tuberculosis (TB) is rare. The risk of occurrence of multifocal TB is increasing in immunocompromised patients. Cutaneous entanglement in multifocal TB occurs more infrequently, and its association with multifocal TB is scarcely reported. The aim of this case report was to show a rare case of lupus vulgaris accompanied by multifocal skeletal TB (vertebrae and knee joint) in a 15-year-old girl. There were kyphosis and deformities seen on physical examination. Dermatological status showed an erythematous plaque on the cheek and an erythematous nodule on the index finger of the left hand. The additional examinations such as bacteriological, histopathological, and polymerase chain reaction (PCR) examinations supported the diagnosis of cutaneous TB. Radiographic and PCR examinations confirmed the diagnosis of multiple skeletal TB. Mycobacterium tuberculosis was obtained from GeneXpert® examination of the skin lesion on the right cheek and synovial fluid of the knee. Therefore, multifocal TB could be confirmed in this patient. The uncommonness of the skin involvement in TB infection should warn the clinicians to commit every possible examination to detect the TB involvement in other organs, especially in immunocompromised children with suspected skin lesions.

Coexisting Bacillus Calmette-Guérin-Induced Lupus Vulgaris Involving the Vaccination Site and Lichen Scrofulosorum in an Immunocompetent Boy.

The coexistence of Bacillus Calmette-Guérin (BCG)-induced lupus vulgaris involving the site of vaccination with lichen scrofulosorum is rare. Herein we report a 3-year-old boy who presented with lupus vulgaris at the vaccination site 3 weeks after neonatal BCG vaccination followed by the development of lichen scrofulosorum approximately 2.5 years later. Characteristic clinical morphology, typical histopathology, and positive DNA polymerase chain reaction for Mycobacterium bovis confirmed the clinical diagnosis.

Ulcerated lupus vulgaris at the site of Bacille Calmette-Guérin vaccination.

We report a case of ulcerated lupus vulgaris occurring in 1.5-year-old boy at the Bacille Calmette-Guérin vaccination site within 6 months, which was diagnosed using histology and polymerase chain reaction. The lesion resolved with isoniazid and rifampicin therapy.

[Lupus vulgaris as a complication of pulmonary tuberculosis--case report]. / Gruzlica toczniowa skóry jako powiklanie gruzlicy pluc--opis przypadku.

Tuberculosis is a contagious disease induced by Mycobacterium species, acid-fast bacilli. These are mostly human type--Mycobacterium tuberculosis, less often cattle type--mycobacterium bovis or other: mycobacterium avium, kansasii, marinom, scrofulaceum, heamophilium, gordonae. The infection can affect all organs, but pulmonary tuberculosis is the most common form. The importance of tuberculosis is definitely rising in the context of massive population migrations in regions affected by its higher incidence, increased HIV infections and AIDS development. Cutaneous tuberculosis is a particular tuberculosis form with differentiated clinical picture. Non-typicalness of skin changes and oligobacilleous course of extrapulmonary tuberculosis forms are repeatedly causing difficulties in adequate diagnosis and early treatment. In differential diagnostics of cutaneous tuberculosis one must take leishmaniasis, actinomycosis, leprosy, syphilis and deep mycosis (among others) into consideration. The study is presenting a case of lupus vulgaris as a complication of past pulmonary tuberculosis. In bacteriological diagnostics of skin changes bioptates, no tuberculosis mycobacteria were found. The disease was diagnosed based on specific granulation presence in histopathology test, tuberculin hypersensivity, bacilli DNA presence in polymerase chain reaction (PCR) test and skin changes regression after anti-mycobacterium treatment. According to authors of the study, the described case confirms the usefulness of PCR nucleonic acids amplification test in cutaneous tuberculosis diagnosis.

Bacillus Calmette-Guérin vaccine- induced lupus vulgaris in a child.

Lupus vulgaris (LV) is a rare form of cutaneous mycobacterial infection in children. Most cases follow hematogenous or lymphatic seeding, and more rarely from exposure to bacillus Calmette- Guérin (BCG) vaccine. We report a child that received BCG vaccination and developed LV 2 months later.

Lupus vulgaris at the site of BCG vaccination: report of three cases.

Lupus vulgaris (LV) is a rare complication of the bacille Calmette-Guérin (BCG) vaccination, and about 65 cases of inoculation tuberculosis resembling LV have been reported in the literature. We report three cases of LV, developing many years later at the inoculation site of BCG vaccine. All three cases had a single BCG vaccination, with a LV lesion at or in the vicinity of the vaccination site, a strong positive Mantoux test, noncaseating granuloma histologically, and two of the patients had a positive PCR result for mycobacterial complex. One of the patients had an unusually delayed appearance of the LV lesion, after an interval of about 17 years, and another case was remarkable because of the large size of the lesion (210 x 110 mm).

[Lupus vulgaris manifestation as a destructive nose and facial tumor]. / Lupus vulgaris als destruierender Nasen- und Gesichtstumor.

Lupus vulgaris is the most frequent manifestation of cutaneous tuberculosis, but in Europe it is limited to isolated cases. Mainly immunocompetent individuals are affected by this result of an endogenous reinfection on a lymphogenous-less frequently hematogenous-pathway. Lupus vulgaris has been observed to develop in more than 50% of all patients who already suffer from other manifestations of tuberculosis. The development of a squamous cell carcinoma in the lupus vulgaris is a rare complication; therefore, lupus vulgaris is deemed a facultative precancerosis.A 68-year-old female Serbo-Croatian patient presented with an extensive ulcerative nose and facial tumor. Her anamnesis included a squamous cell carcinoma of the nose that had been excised alio loco 3 years before. Further examinations revealed enlarged cervical lymphoma on both sides, and pulmonary metastases were also suspected. The tumor biopsy revealed a necrotic, granulomatous inflammation. No acid-fast rods were seen on Ziehl-Neelsen stain. The tuberculous origin of this ulcerative skin tumor-the lupus vulgaris-as an endogenous reinfection of pulmonary tuberculosis manifestation was confirmed by the detection of Mycobacterium tuberculosis DNA in polymerase chain reaction and the growth of Mycobacterium tuberculosis colonies in the bacterial culture (skin biopsy and bronchial secretion). The skin tumor as well as the pulmonary manifestation were successfully treated with combined tuberculostatic therapy and showed a dramatic response within 3 months.

Bacillus Calmette-Guérin vaccine-induced lupus vulgaris in a child adopted from China.

Lupus vulgaris is a rare form of cutaneous mycobacterial infection that can occur from mycobacterial exposure and even more rarely from exposure to the Bacillus Calmette-Guérin vaccine. We report a child who received this vaccination in China and then developed lupus vulgaris shortly after being adopted in the United States. After histopathologic confirmation, the infection was successfully treated with a combination of antibiotics. This occurrence demonstrates the need for heightened surveillance of Bacillus Calmette-Guérin vaccine-induced lupus vulgaris, given the increasing numbers of overseas adoptions taking place in the United States. To our knowledge, this is the first reported instance of Bacillus Calmette-Guérin immunization-induced lupus vulgaris in the United States.

Environment versus genetics in autoimmunity: a geneticist's perspective.

Both environmental as well as genetic factors have been documented to contribute to autoimmunity, as is evident from the accompanying articles in this journal. This raises an important question - which of these two elements plays a more important role in the genesis of autoimmunity? This report encapsulates a geneticist's perspective on this interesting question. Though lupus is used as an example in this essay, the proposed model is envisaged to operate in other autoimmune diseases as well.

Kinetics of human B cell behavior and amplification of proliferative responses following stimulation with IL-21.

Although recent studies indicated that IL-21 is an important regulator of human B cell activation, detailed comparison of the effects of IL-21 on distinct B cell subsets have not been performed. Our studies revealed that IL-21R is expressed by naive and germinal center B cells, but not memory or plasma cells. IL-21R was increased on naive and memory B cells following in vitro activation. Investigation into the kinetics and magnitude of responses of human B cells to IL-21 revealed that IL-21 potently augmented proliferation of CD40L-stimulated neonatal, splenic naive, and memory and tonsil germinal center B cells. This response exceeded that induced by IL-4, IL-10, and IL-13, cytokines that also induce B cell proliferation. Remarkably, CD40L/IL-21-stimulated naive B cells underwent the same number of divisions as memory cells and exhibited a greater enhancement in their response compared with CD40L alone than memory B cells. Therefore, IL-21 is a powerful growth factor for naive B cells. This may result from the higher expression of IL-21R on naive, compared with memory, B cells. Stimulation of human B cells with CD40L/IL-21 also induced IL-10 production and activation of STAT3. We propose that IL-21 may have therapeutic application in conditions of immunodeficiency where it could expand naive B cells, the predominant B cell subset in such patients. Conversely, because IL-21 is increased in murine models of lupus, dysregulated IL-21 production may contribute to perturbed B cell homeostasis observed in systemic lupus erythematosus. Thus, antagonizing IL-21 may be a novel strategy for treating Ab-mediated autoimmune diseases.

[A case report of childhood systemic lupus erythematosus complicated with lupus cystitis].

The patient was a 13-year-old girl. In August 2000, she presented with a fever, together with diarrhea, vomiting, arthralgia, nasal bleeding and malaise, and was examined by another physician. Because her platelet count was low, and there were positive reactions for anti-nuclear antibodies, anti-DNA antibodies and platelet-associated IgG, idiopathic thrombopenic purpura, and systemic lupus erythematosus (SLE) was suspected. From January 2001, when she caught measles, she reported abdominal pain, and urinalysis indicated urinary protein and occult blood, and the left kidney was found hydronephrotic. At the same time left ureter stenosis and dilatation were demonstrated. Symptoms were disappeared by hydration and treatment with NSAIDs, but 2 months later fever and erythematous patches seen on both cheeks led to the proper diagnosis of SLE, and she was admitted to our hospital. Intravenous pyelography revealed hydronephrosis on left kidney, constriction and dilatation of the left ureter, and intracystic endoscopy showed erythema at the orifice of the left ureter. The pathological examination indicated the presence of vasculitis, and finally lupus cystitis was diagnosed. Intravenous cyclophosphamide (IVCY)-pulse therapy was introduced to a total of 8 times over the period of a year, and maintenance therapy with predonisolone and azathioprin was also used. After completion of the IVCY-pulse therapy, the hydronephrosis and constriction of the ureter were disappeared. No side effects of IVCY-pulses were observed, and the patient is now in remission. We reported a case of childhood SLE complicated with lupus cystitis and successfully treated by IVCY-pulse therapy and maintenance predonisolone and azathioprin.