Lupus Vulgaris at the site of Bacillus Calmette – Guerin vaccine injection, Case Report
Khalifa Mohie aldeen Abd Alzaher Mohamed *,
Awadallah Mohamed Fawzi. **
Alian Hassan Abd Elbary ***
* Department of the chest, Mataria teaching hospital, General Organization for Teaching Hospital and Institute, Cairo, Egypt.
**Department of the chest, Faculty of Medicine AL-Azhar University, Damietta Egypt
***Department of Dermatology and Venereology Mataria Teaching Hospital, General Organization for Teaching Hospital and Institute, Cairo, Egypt.
khalifa Mohie Aldeen, Department of Chest Disease, Mataria Teaching Hospital, General Organization for Teaching Hospital and Institute, Cairo, Egypt.
E-mail: [email protected]:
Introduction: Lupus vulgaris is known as a rare form of cutaneous tuberculosis as a complication of BCG vaccination.
Clinical presentation: Male patient with painless, slowly progressive cutaneous abnormality on the left deltoid area at the site of BCG vaccination injection extended to the shoulder and chest wall. Diagnosis of post BCG lupus vulgaris made by histopathological examination of the biopsy taken from the edge of the lesion and confirmed by polymerase chain reaction. The patient was started on antituberculous drugs with good response.
Conclusions: Histopathological and PCR studies are useful methods for diagnosis of Lupus vulgaris in add response to specific ant-tuberculosis treatment. The diagnosis and treatment of our patient were delayed to the age of 27 years. Many providers are unfamiliar with its clinical manifestations of BCG-induced lupus vulgaris. So we recommended that all healthcare provider should be aware of this rare clinical condition and treat it appropriately as early as possible.
Key-words: Lupus Vulgaris, BCG vaccination, Complication, Tuberculosis
Introduction: Cutaneous tuberculosis is a rare form of extrapulmonary tuberculosis, which manifests in 8.4–13.7% of all tuberculosis1, most cases developed follow hematogenous or lymphatic seeding and more rarely from exposure to Bacillus Calmette-Guérin(BCG)vaccine 2.BCG vaccine-induced Lupus vulgaris risk is estimated at 5/1,000,0003. Local adverse reactions post BCG vaccination include pustule formation with regional lymphadenopathy and sinus formation, more serious local reactions include ulceration at the vaccination site. These manifestations might occur up to 5 months after vaccination and could persist for several weeks 4.
Case Presentation: On January 2018, A twenty-seven-year-old male patient arrived at our hospital for clinical evaluation and medical advice where he had a 21 years history of a cutaneous abnormality characterized by extending and progressive erythematous plaques with an elevated scaly surface on his left shoulder and to the anterior aspect of left hemithorax. His family history was negative for tuberculosis. According to his medical history, the patient received BCG vaccination on his left deltoid area, the lesion was observed at 6 years old age and started as a small papule at the injection site of BCG that enlarged gradually and changing to an erythematous, edematous plaque, when he was 15 years old, and since then the lesion had been slowly enlarging from several centimeters in diameter to 15-20 cm in different diameter in his twenties age. He denied fevers, night sweats, weight loss, cough, and dyspnea. Clinical examination revealed a 20-25 cm in different diameter cutaneous lesion with reddish-brown nodules that showed apple-jelly appearance on diascopy(Figure 1), these lesions were neither painful nor itchy. There was no axillary or cervical lymphadenopathy. Routine laboratory studies including complete blood count, chemistry panel serum (urea, creatinine, uric acid, bilirubin total and direct, SGOT, SGPT, alkaline phosphatase, total protein, albumin, sodium, potassium were within normal range. But ESR was (90 mm)in 1st hr, A tissue culture did not yield a Mycobacterium species. Smear culture from the lesion revealed neither fungi nor skin contamination. Serology for HIV was negative. Culture and Gram stain were negative for bacteria. Intradermal Mantoux (5 TU PPD) was positive (20 mm) at the end of 48 hours, the Skin biopsy was taken from the edge of the lesion by Ribble Dermal Biopsy Punch size #3.00 MM(Figure 2)and histopathological examination was done. It was reported as a chronic granulomatous inflammation suggestive of tuberculosis. The biopsy specimen contained multiple granulomas composed of epithelial cells and Langan’s giant cells; areas of necrosis and dense lymphocytic infiltrate. No acid-fast bacillus was detected in Ziehl-Neelson staining. However, PCR of tissue biopsied detected M. tuberculosis complex. Plain X-ray chest anteroposterior and right lateral were apparently normal. A diagnosis of BCG-induced lupus vulgaris was made. Therefore, the patient was started antituberculous drugs in the form of Isoniazid 300 mg/day, Rifampin 600 mg/day, Ethambutol 1500 mg/day, pyrazinamide 1500 mg/day, and vitamin B 6 for two months. The skin lesions had regressed within eight weeks of antituberculous therapy (Figure 3) followed by continuation phase, for the next four months with Isoniazid 300 mg/day and Rifampin 600 mg/day with good response and lesions regress with residual skin atrophy (Figure 4).
BCG vaccination has been used in Europe to prevent tuberculosis in humans since 19215, Since that time no other method of preventing tuberculosis in children has been known. The BCG vaccination is still regarded as safe and effective6,but had some complications include: Local reaction such as axillary or cervical lymphadenopathy, local subcutaneous abscess, and keloids, skin lesions distinct from the BCG vaccination site such as TB chancre, lupus vulgaris, scrofuloderma, papulonecrotic tuberculids, and BCG-oasis in immunocompromising diseases7.
Lupus vulgaris is usually the result of dissemination from an endogenous focus during a period of lowered resistance and mycobacterium tuberculous bacillemia in a previously sensitized host with a strongly positive delayed hypersensitivity to tuberculin 5-6. The intensity and duration of the local reaction depend on the depth of penetration of the multiple puncture device and individual variations in patients’ tissue reactions 2.
In our patient’s diagnosis and treatment were delayed to the age of 27 may be related to the patient not seeking care or having limited access to care, misdiagnosis can also significantly delay appropriate treatment. Many providers are unfamiliar with its clinical manifestations; Rather than the hidden location, overlooking changes in the appearance of existing or omitting new lesions main reasons for the patient-related delay. Our patient’s lesion was noticed at six years old, but other studies recorded that, Lupus vulgaris develops few weeks to 8 years after BCG vaccination and mean duration for BCG-induced Lupus vulgaris is one year3. The size of the lesion in our patient was 20×25 cm in different diameter, on the other hand, the largest reported size of Lupus vulgaris lesion is 60×45 cm8. Usually, Mycobacterium is not found in culture because Lupus vulgaris is a paucibacillary form of TB9. Similar to in our patient, We did not detect the Mycobacterium in acid-fast staining and culture and this finding is supported by Steidl et al10. Diagnosis of our patient depends on the history is taken, the clinical assessment supported by the positivity of Tuberculin Skin Test and confirmed by, histopathological study PCR and response to therapy these diagnostic steppes is striate forward as similar to other recorded cases 8,9,10. Awareness of varied clinical presentations with a high index of clinical suspicion of cutaneous tuberculosis, is the key to early diagnosis and treatment, thus reducing the morbidity1.
Conclusion and recommendation: Lupus vulgaris is a rare complication of the post-BCG vaccine. Lupus vulgaris is a paucibacillary form of TB. Therefore, acid-fast bacilli are not detected. The histopathological study and PCR is a useful method for confirmation of the diagnosis in adds response to specific ant-tuberculosis treatment. Many providers are unfamiliar with its clinical manifestations of Post immunization Lupus vulgaris. So we recommended that dermatologists, pediatricians, pulmonologists, primary health care provider and other specialists should be aware of this rare presentation and treat it appropriately early as possible.
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The authors thank the patient for cooperation and agreement the publication.
Figure 1: Clinical photograph showing apple-jelly lesion at the BCG vaccination site expanding to the anterior chest wall and left shoulder, lesion initially before antituberculosis treatment.
Figure 2: Ribble Dermal Biopsy Punch size #3.00 MM
Figure 3: Clinical photograph showing the lesion, two months later with antituberculosis treatment
Figure 4: Clinical photograph showing healed lesion with atrophic scarring over left shoulder after 6 months of antituberculous treatment.