|Year : 2019 | Volume
| Issue : 2 | Page : 205-207
A rare case of multiple lupus vulgaris in a multifocal tuberculosis pediatric patient
Hendra Gunawan, Gynaecia Lamsu, Pati Aji Achdiat, Oki Suwarsa, Reti Hindritiani
Department of Dermatology and Venereology, Faculty of Medicine, Padjadjaran University – Dr. Hasan Sadikin General Hospital, Bandung, Indonesia
|Date of Web Publication||14-Jun-2019|
Department of Dermatology and Venereology, Faculty of Medicine, Universitas Padjadjaran – Dr. Hasan Sadikin General Hospital, Bandung
Source of Support: None, Conflict of Interest: None
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.
Keywords: Cutaneous tuberculosis, lupus vulgaris, multifocal tuberculosis
|How to cite this article:|
Gunawan H, Lamsu G, Achdiat PA, Suwarsa O, Hindritiani R. A rare case of multiple lupus vulgaris in a multifocal tuberculosis pediatric patient. Int J Mycobacteriol 2019;8:205-7
|How to cite this URL:|
Gunawan H, Lamsu G, Achdiat PA, Suwarsa O, Hindritiani R. A rare case of multiple lupus vulgaris in a multifocal tuberculosis pediatric patient. Int J Mycobacteriol [serial online] 2019 [cited 2020 Feb 29];8:205-7. Available from: http://www.ijmyco.org/text.asp?2019/8/2/205/260371
| Introduction|| |
Tuberculosis (TB) can affect many organs or systems of the human body and its clinical manifestations in each system are diverse. It is still remains a major health problem worldwide. Globally, TB is the second leading cause of death from an infectious disease. Cutaneous TB, although rare, is a form of extrapulmonary TB that accounts for only 1%–2% of total TB cases., The disease is usually limited to the skin but can be multifocal, especially in immunocompromised patients. Multifocal TB is uncommon and occurs, especially in immunocompromised patients, and the association with skin involvement is even rarer.
Among a large variety of cutaneous TB, lupus vulgaris (LV) is the most common morphological variant, accounting for approximately 59% cases of cutaneous TB. LV is a form of cutaneous TB with varying clinical manifestations, solitary form, and asymptomatic., The lesions of the disease can clinically arise in multiple forms in children and immunocompromised patients. LV is diagnosed based on a combination of clinical presentation, histopathological examination, and bacterial culture results. A majority of cases of LV, both confirmed and presumptive, have been treated with anti-TB drugs with good response.
The current study is a case report of a 15-year-old girl. She presented with multiple LV lesions and multifocal skeletal TB. This case proves cutaneous TB in children is often preceded by systemic TB infection. This report will be helpful toward planning more awareness about cutaneous TB in children for health-care providers to consider the screening of internal organs involvement leading to early diagnosis and preventing lethal complications.
| Case Report|| |
A 15-year-old girl was consulted from the Department of Child Health with multiple skin lesions. There was an erythematous plaque on the right cheek and an ulcerated nodule on the index finger of the left hand. Both of the lesions presented without pruritus or tenderness. The skin lesion on the right cheek appeared since 1 month before consultation as a button-sized erythematous plaque that enlarged to the coin-sized lesion. The lesion on the left hand appeared since 7 months before consultation as a pea-sized nodule that became ulcerated which eventually healed. The lesion always occurs periodically and never been completely healed. The patient was admitted to the hospital due to the complaint of her inability to move because of the weakness of both lower extremities and pain on her vertebrae that felt since 5 months before hospitalized. The diagnosis of spondylitis TB and arthritis TB was already suspected. There were no history of vaccination and personal TB infection. However, the patient's younger sister is currently on therapy for meningitis TB. The father of the patient has passed with the unknown cause but had a history of having the same symptoms of weakness on both lower extremities. Physical examination showed one well-defined erythematous plaque [Figure 1]a, around 3 cm in diameter on the right cheek and a crusted nodule on the left index finger [Figure 1]b. There were also multiple inguinal lymphadenopathies. On the diascopy test, “apple jelly” sign was negative.
|Figure 1: Clinical manifestations of lupus vulgaris (a) erythematous plaque on the right cheek (black circle), (b) erythematous nodule on the index finger of the left hand with crusted surface (red circle)|
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Further investigations for establishing the diagnosis were performed. Tuberculin test was already being done before consultation by the Department of Child Health, and the result was positive. The bacterial examination from the ulcer on the left hand showed acid-fast bacilli (AFB) [Figure 2]a. Biopsy procedure was performed from the erythematous plaque on the cheek for histopathological, bacterial culture, polymerase chain reaction (PCR), radiographic, and GeneXpert® examinations. Histopathological examination showed Datia Langhans cell that supported the diagnosis of LV [Figure 2]b. Bacterial culture revealed no growth of Mycobacterium tuberculosis (M. tuberculosis). PCR examinations revealed a positive result of the bacteria. The radiographic examination result of the vertebrae revealed spondylitis TB. The patient also underwent a surgery procedure for collecting synovial fluid from the knees for further GeneXpert® examination. M. tuberculosis was obtained from GeneXpert® examination of the right cheek and synovial fluid of the knee. The patient was kept on oral anti-tuberculosis therapy (OAT) based on the National TB Control Programme Guideline Indonesia 2014. Response to therapy was noted by the change of the skin lesions; some parts of the erythematous plaque became erythematous macule, and the lesion on the left index finger became hyperpigmented plaque.
|Figure 2: (a) Acid-fast bacilli (black arrow) from the bacterial examination of the index finger of the left hand, (b) Langhans giant cell (red circle) on the histopathological examination from the erythematous plaque of the right cheek (H and E, ×400)|
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| Discussion|| |
Extrapulmonary TB occurs in 10% of all TB cases and may result from exogenous inoculation, spreading from an adjacent focus, or a hematogenous spread from a distant focus. LV is a chronic progressive form of cutaneous TB occurring in patients with moderate-to-high degree of immunity, originating from an underlying TB (endogenous) focus or exogenous inoculation. Most of the times, LV occur in solitary form, but in children and immunocompromised patients, it can arise in multiple forms. There are five typical forms of LV such as plaque form, ulcerative/mutilating, vegetative, tumor like, and the papular/nodular form. LV can occur simultaneously with TB infection in other organs, 40% of cases with lymphadenitis TB, 30% of cases with scrofuloderma, and 10%–20% of cases with pulmonary or skeletal TB. The patient in this report was diagnosed with spondylitis TB and arthritis TB based on radiographic examination of the vertebrae and PCR from synovial fluid of the knee.
Multifocal TB is defined as the presence of two or more infections caused by M. tuberculosis in extrapulmonary sites, with or without pulmonary involvement. The suspicion of multifocal TB should prompt researchers to look for an underlying immunosuppressed conditions, because it is confined mostly to immunocompromised patients. This disease can affect any visceral organs by two suggested mechanisms, such as hematogenous and lymphogenous. Three types of multifocal TB can be differentiated based on the organ involvement, consist of multifocal skeletal TB, multifocal intestinal TB, and multifocal systemic TB., The concomitant involvement of skin and skeletal TB is a very rare condition. Kivanç-Altunay et al. reported that 61% of cutaneous TB cases had pulmonary involvement, whereas no skeletal TB was encountered. Kumar et al. reported that four patients out of 75 with cutaneous TB had skeletal TB.
The diagnosis of LV is established by absolute and relative criteria. The absolute criteria is the finding of M. tuberculosis from bacterial culture, direct inoculation, or PCR. Relative criteria consists of proper related history and clinical findings, active TB infection in other organs, positive bacteriological examination, supported histopathological result, positive tuberculin test, and good outcome on OAT treatment., Negative results from those examinations does not exclude the diagnosis, because it is not uncommon that the diagnosis of LV can be obtained only based on clinical and pathological correlation. A proper diagnosis of cutaneous TB requires a good correlation of clinical findings and diagnostic testings, such as the finding of AFB on smears, cultures, PCR, and histopathological examinations. The diagnosis of LV in this patient was achieved by correlating history taking, physical findings, and additional examinations such as histopathological and PCR examinations, also good therapeutic response. The establishment of multifocal TB is often delayed due to the nonspecific sign and symptoms,, as well as confirming the diagnosis of skeletal TB. Skeletal TB can be verified by several examinations such as conventional radiography, magnetic resonance imaging, and computed tomography scans. It should be noted that these procedures often show similar descriptive images that can also be seen in other diseases such as malignancy. Therefore, the definitive diagnosis is based on histopathological findings., This patient underwent various examinations. Based on the clinical scenario, histopathological, and PCR examinations, the diagnosis of the skin lesions can be confirmed as LV. The skeletal TB diagnosis in this patient was confirmed by conventional radiography of the vertebrae and a positive result on GeneXpert® examination taken from synovial fluid of both knees. Therefore, the patient, in this case, was diagnosed as LV with multifocal skeletal TB.
The management of all cutaneous TB is similar to pulmonary TB as well as extrapulmonary TB. According to the National TB Control Programme Indonesia 2014, extrapulmonary TB should be treated with category 1 OAT regimen. Multifocal TB is treated with the same regimen of pulmonary TB only with the longer duration. Skeletal TB is treated with OAT for 12–18 months' duration. Most of the cutaneous TB cases present good responses to OAT. Until this report is written, the patient in this report is still undergoes a 7th month treatment with the category 1 OAT drugs and is planned to have the therapy for 12 months.
| Conclusion|| |
We report this case considering the occurrence of multiple LV lesions alongside with systemic organ involvement in an immunocompromised child. Cutaneous TB in children continues to be an important cause of morbidity, and there is a high likelihood of internal involvement., Therefore, it should be a prime of importance to put LV as one of the differential diagnosis on any chronic skin lesions and the detection of TB infection in other visceral organs should be vital if there is suspected LV lesions in children.
Consent and ethical clearance
Written informed consent was obtained from the patient for publication of this case report and accompanying images.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understands that name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
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[Figure 1], [Figure 2]