Introduction:

Extrapulmonary tuberculosis (TB) accounts for about 15% of all TB infections.[1] Lupus vulgaris (LV) is the most common morphological variant of cutaneous tuberculosis, accounting for 75% of the cases, and was first described by Erasmus Wilson in 1865.[2] Nasal tuberculosis accounts for about 6.7% of extrapulmonary tuberculosis. The involvement of lacrimal drainage is even rarer.[3] It was first reported by the Italian anatomist Giovanni Battista Morgagni. The coexistence of LV with sinonasal tuberculosis and dacrocystitis is very rare and is infrequently reported.

Case Report

A 72 year old female presented to the ophthalmology outpatient department (OPD) with complaints of epiphora and purulent discharge from the right eye for the past 2 years. On ophthalmic examination, her right nasolacrimal duct was partially obstructed. There was a history of surgery done for cataract for both eyes. She also complained of nasal blockage, for which she was referred to the ENT department. On diagnostic nasal endoscopy, polypoidal changes were noted in the bilateral middle meatus near the maxilla. She also had multiple asymptomatic skin lesions over the right upper eyelid and right arm for the past 1 year, for which she was referred to our Dermatology OPD. There was no history of trauma. There was no prior history of pulmonary tuberculosis. She had been a known case of type 2 diabetes mellitus and hypertension for the past 10 years on regular treatment. On cutaneous examination, an infiltrated erythematous plaque over the right upper eyelid was noted, measuring size 2×1 cm. There were also multiple erythematous plaques noted over the right arm extending to the periphery with indurated margins and central scarring, with the size ranging from minimum 4×2 cm to maximum 7×5 cm. (Figure 1a & b)

Figure 1: an infiltrated plaque in the right upper eyelid (a) and multiple atrophic plaques with erythematous indurated border in the right upper arm (b) before starting ATT	Figure 2: On diascopy, apple jelly nodules were noted over the plaque

On diascopy, apple jelly nodules were noted, suggesting LV. (Figure 2) The tuberculin skin test carried out with 0.1 ml of purified protein derivative showed a positive test with an induration of 14 mm at 72 hrs. (Figure 3) Histopathological examination of the skin lesion revealed multiple non-caseating epithelioid cell granulomas with Langhan's giant cells located in the superficial and mid dermis. (Figure 4)

Figure 3: Mantoux test showing an induration of 14 mm over the left upper forearm	Figure 4: Histopathology of skin lesion showing the presence of non caseating granuloma composed of epithelioid histiocytes, Langhan's type giant cells and lymphocytic infiltrates in the dermis.

Her chest X-ray was found to be normal. CT paranasal sinuses (PNS) revealed mucosal thickening in the bilateral frontal, bilateral anterior, middle and right ethmoidal air cells with a soft tissue density in the medial canthus of right eye, suggesting the involvement of sinuses and dacrocystitis. (Figure 5) We finalised the diagnosis as LV with sinonasal TB and dacryocystitis after a complete workup and referred the patient to a nearby government center for starting on ATT. After four months of treatment, her complaint of epiphora resolved completely, and skin lesions over her right upper eyelid and right arm were notably decreasing with a reduction in erythema and induration. (Figure 6)

Figure 5: CT PNS showing bilateral frontal sinuses mucosal thickening (a), bilateral anterior, middle and right posterior ethmoidal sinuses mucosal thickening (b) and soft tissue density lesion in the medial canthus of right eye (c)	Figure 6: resolution of lesions over right upper eyelid and right upper arm after 4 months of starting ATT

Discussion

LV can be acquired by exogenous inoculation, contiguous extension of the disease from the underlying affected tissue, or by hematogenous or lymphatic spread.[4] It can also occur due to the reactivation of tubercular foci. The common sites affected are the face in western countries and the buttocks and extremities in India.[5] It is predominant in females than in males at 2-3:1 ratio in Philippines, whereas in India it is more common in males with a male to female ratio of 6-8:1. It starts as a soft brownish red papule or nodule that gradually expands by involution in one area with expansion in another to form a well defined skin coloured or erythematous plaque with an "apple jelly nodular" appearance on diascopy, as observed in our case. Bhutto et al[6] described four major types of LV. They are psoriasiform, nodular, papular, and erythematous. It has the tendency to spread and cause the destruction of surrounding structures. The nasal, buccal, and conjunctival mucosa can be involved primarily or by extension. Various malignancies have been reported to occur in long standing cases of LV, which include squamous cell carcinoma and basal cell carcinoma.[7,8]

The tuberculosis of nose usually occurs secondary to pulmonary tuberculosis through a contagious, hematogenous, or lymphatic route and is frequently associated with immunocompromised states, diabetes, and HIV infection. Primary sinonasal tuberculosis is extremely rare because the nose and paranasal sinuses are most resistant to tuberculous invasion due to their unique epithelial architecture and bactericidal secretion.[4] It is more common in females than in males. Among the paranasal sinuses, unilateral maxillary sinus is the most common presentation. In contrast, bilateral frontal and ethmoidal air cells were involved in the present case. The patients can have nasal obstruction, blood stained nasal discharge, epistaxis, crusting, dryness of the nose and throat, epiphora, postnasal discharge, and blurred vision. It usually affects the nasal septum, anterior segment of inferior turbinate, PNS, choana, nasopharynx, orbit, and cranial cavity. Sinonasal tuberculosis often presents with considerable clinical variation, like LV, which is the most common presentation, while it may present in a non-ulcerated form mimicking septal swelling.

TB of orbit is a very uncommon occurrence. It is acquired by hematogenous spread, direct extension from an adjoining site like sinus or lacrimal gland, or by incidental conjunctival contamination with tubercle bacilli and subsequent passage of the same to the lacrimal sac with tears.[9] Dacrocystitis is generally seen as a result of nasolacrimal duct obstruction, where inadequate drainage of tears leads to swelling of lacrimal sac and watering of the eyes. Having discussed about LV, sinonasal TB, and dacrocystitis, it has been clarified that they can present as a separate entity or in a combined form as reported in the previous studies (Table 1). In the present case, LV of eyelid would have occurred secondary to lacrimal sac TB and subsequently spread to involve the right arm.

The initial presentation of TB dacrocystitis is usually nonspecific, with complaints like watering eyes as in our case, which could be easily missed out. [10,11] Though epiphora was the initial presentation, subsequent findings such as CT-PNS suggesting the involvement of sinuses and dacrocystitis, the classic cutaneous findings of LV with characteristic apple jelly nodules, histopathological examination of skin tissue showing noncaseating Langhan's giant cell granuloma, and a positive tuberculin test helped us to diagnose the patient as having LV coexisting with sinonasal TB and dacrocystitis.

Conclusion

We report this rare occurrence of LV with sinonasal tuberculosis and tuberculous dacrocystitis in order to raise awareness regarding this unusual association among the physicians. Tuberculous dacryocystitis has to be considered in any patient with watering or purulent discharge from the eyes, especially when coexistent with lupus vulgaris.

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