A case of tuberculous dacryoadenitis with review of literature

Sumeet Lahane; Sheetal Munde; Ramanna Chalvadi; Dhananjay Prajapati; Ragini Parekh; Tatyarao Lahane

doi:10.4103/ijo.IJO_919_21

Users Online: 745

CASE REPORT

Year : 2022 | Volume : 2 | Issue : 1 | Page : 220-222

A case of tuberculous dacryoadenitis with review of literature

Sumeet Lahane, Sheetal Munde, Ramanna Chalvadi, Dhananjay Prajapati, Ragini Parekh, Tatyarao Lahane
Grant Government Medical College and Sir JJ Group of Hospitals, Mumbai, Maharashtra, India

Date of Submission	18-Apr-2021
Date of Acceptance	13-Jul-2021
Date of Web Publication	07-Jan-2022

Correspondence Address:
Dr. Sumeet Lahane
Grant Government Medical College and Sir JJ Group of Hospitals, Mohammed Ali Road, Byculla, Mumbai, Maharashtra
India

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ijo.IJO_919_21

Abstract

Lacrimal gland inflammation is commonly due to viral infection, autoimmune, and idiopathic inflammatory disease. Orbital and adnexal tuberculosis (OTB) is a rare entity even in the TB endemic country of India. TB dacryoadenitis is one of the presentations in the OTB spectrum, with less than 25 cases in the literature. We report the case of a 14-year-old female primarily presented with unilateral dacryoadenitis and cold abscess at the lower lumbar region. Biopsy of lacrimal gland confirmed on histopathology as tuberculosis. Systemic investigation revealed pulmonary and extrapulmonary TB. She was treated with systemic category I anti-tubercular treatment resulting in complete regression of disease.

Keywords: Dacryoadenitis, lacrimal gland mass, ocular tuberculosis, tuberculous dacryoadenitis

How to cite this article:
Lahane S, Munde S, Chalvadi R, Prajapati D, Parekh R, Lahane T. A case of tuberculous dacryoadenitis with review of literature. Indian J Ophthalmol Case Rep 2022;2:220-2

How to cite this URL:
Lahane S, Munde S, Chalvadi R, Prajapati D, Parekh R, Lahane T. A case of tuberculous dacryoadenitis with review of literature. Indian J Ophthalmol Case Rep [serial online] 2022 [cited 2022 Jan 21];2:220-2. Available from: https://www.ijoreports.in/text.asp?2022/2/1/220/334991

Inflammation of the main or accessory lacrimal gland is known as dacryoadenitis. It could present as an acute or chronic disease and is commonly unilateral. Epstein Barr Virus is the most common cause of dacryoadenitis.^[1] Adenovirus, herpes simplex, varicella-zoster, mumps, rhinovirus, and Cytomegalovirus are also reported etiological agents. Along with infective agents as S. aureus, S. pneumoniae, Nocardia, few autoimmune diseases such as non-specific inflammatory orbital disease, IgG4 related disease, or Sarcoidosis can also involve the lacrimal gland. TB dacryoadenitis (TBD) was first reported by Abadie (1881).^[2] Since then, well-documented cases in the literature are less than 25, with more than 50% of Indian-origin patients. We report a case with a primary presentation of dacryoadenitis and later diagnosed as TB.

Case Report

A 14-year-old female patient presented with complaints of swelling over the left upper eyelid for one month which was painless but gradually increasing in size. Visual acuity in both eyes was 6/6, N6. Left eye (LE) anterior segment was normal, with a palpable firm, cystic swelling, and well-defined margins at supero-temporal aspect involving lacrimal gland [Figure 1]a. Right eye (RE) was normal. Schirmers I test in RE and LE was 15 and 7 mm, respectively, at 5 minutes, suggestive of reduced LE lacrimal gland function.

Figure 1: (a) Clinical picture showing left eye upper eyelid swelling in supero-temporal region; (b) Clinical picture showing cold abscess over the lower thoracic region; (c) Computed tomography of the orbit (axial section); (d) Computed tomography of the orbit (coronal section); (e) Histopathology showing Langhans giant cell (black arrow) and caseous necrosis with epithelioid cells, lymphocytes; and (f) HRCT lungs showing tree-in-bud appearance

Click here to view

On systemic examination, soft, cystic, non-tender, well-defined swelling was noted in the right paravertebral area of the lower thoracic region and over the scalp in the occipital region measuring 8 × 5 × 2 cm³ and 1 × 1 × 1 cm³, respectively [Figure 1]b. The patient's sibling had succumbed due to TB a few months back. Computed tomography of orbit revealed well-defined hypodense cystic mass located at the lateral wall of left orbit involving the lacrimal gland without any bony erosion or periostitis [Figure 1]c and [Figure 1]d.{Figure 1}

Excision biopsy on histopathology (HPE) showed fibro-collagenous tissue with multiple epithelioid cell granuloma and Langerhans type of giant cells, areas of caseous necrosis, and chronic inflammatory cellular infiltrate comprising lymphocytes and plasma cells suggestive of TB [Figure 1]e.

High-resolution computed tomography (HRCT) of the thorax revealed multiple centrilobular nodules forming tree-in-bud opacities in the superior segment of the right lower and middle lobe of the lung associated with patchy areas of consolidation [Figure 1]f. The diagnosis of tuberculous dacryoadenitis was made based on a positive Gene Xpert report for Mycobacterium TB, HRCT thorax findings of tuberculosis, cold abscess over the back region, and HPE. The patient was started on an anti-tubercular regimen – category I Anti-Koch's treatment (AKT). Complete resolution of disease entity was noted without recurrence at 3-month follow-up post-AKT.

Discussion

The prevalence of tuberculosis in India is estimated to be 5.05 per thousand; the prevalence of smear-positive cases is 2.27 per thousand, and the average annual incidence of smear-positive cases is 84 per 1,00,000 annually. The incidence of ocular tuberculosis varies from 1.4% to 7.8% according to the literature.^[3] Two major review studies of Helm et al.^[1] and Madge^[4] report 43 and 79 cases of OTB, respectively, with only 8 cases of lacrimal gland involvement.^[4],[5] We found a total of 23 cases of dacryoadenitis in the literature [Table 1].^{[5],[6],[7],[8],[9],[10]}

Table 1: TB dacryoadenitis reported cases

Click here to view

The mean age of presentation was 28.35 (±18.89) years. Out of the 23 reported cases, 3 (13%) were male and 20 (86.95%) were females. Three patients were in the pediatric age group of <12 years. Seven cases had bilateral involvement and 16 were unilateral; 13 (56.5%) patients were of Indian origin and Syria, Somalia, Scottish, Senegal, Switzerland, Nepal, Philippines, Tunisia, Istanbul, Egypt had one case each. The most common symptom at the time of presentation was swelling of the upper eyelid. The diagnosis was established by biopsy in 16 (69.56%) patients, fine needle aspiration cytology (FNAC) in 3 (13%), and clinical working diagnosis in 4 (17%) patients [Table 1]. Acid-fast bacilli were found in 3 cases (13%); 7 (30.4%) cases had pulmonary involvement. None of the cases reported recurrence of infection. Though it has been noted that resurgence of TB cases happened post 1980 due to simultaneous occurrence with Human Immunodeficiency Virus (HIV), out of 23 cases of lacrimal gland TB only one HIV-positive case was reported, contradicting the resurgence of TB with HIV in dacryoadenitis. All these patients were treated with anti-tubercular therapy.^{[3],[4],[5],[6],[7],[8],[9],[10],[11]}

Orbital tuberculosis is classified as (i) classical periostitis; (ii) orbital soft tissue tuberculoma or cold abscess, with no bony destruction; (iii) orbital TB with evidence of bony involvement, not categorized as (i); (iv) orbital spread from the paranasal sinuses; and (v) dacryoadenitis by Madge et al.^[4] Etiopathogenesis suggested that tubercular bacilli spread via hematogenous route after rupture of hilar lymph nodes into pulmonary veins or contiguous spread from the neighboring paranasal sinuses. Bacilli invade the lacrimal gland and may remain dormant in intracellular locations till suppressed by systemic and local immunity.^[5]Though the primary focus of TB is suspected to be the respiratory system, lung involvement was noted in only 30% of reported cases, which suggests an alternative route of ocular involvement.

Systemic workup such as Tuberculin skin test (TST), interferon-based immunological test, HRCT chest, erythrocyte sedimentation rate (ESR), complete blood count (CBC), biopsy/FNAC, early start, and completion of AKT is recommended protocol to be followed in OTB cases.

Conclusion

TBD should be considered an important differential for patients presenting with enlargement of the lacrimal gland especially in populations from endemic areas like India. Though biopsy of the lesion helps in definitive diagnosis; history of TB contact, prodromal symptoms, chest Xray/HRCT chest, CT scan orbit, TST, ESR, CBC, ethnicity, and FNAC of lesion combined together are reliable indicators for clinical diagnosis of TBD.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.[21]

References

1.	Helm CJ, Holland GN. Ocular tuberculosis. Surv Ophthalmol 1993;38:229-56.
2.	Abadie C. Tumeurs rares symetriques des paupieres [uncommon symmetrical swelling of the eyelids]. Arch Ophthalmol 1881;1:432-7.
3.	Chakraborty AK. Epidemiology of tuberculosis: Current status in India. Indian J Med Res 2004;120:248-76.
4.	Madge SN, Prabhakaran VC, Shome D, Kim U, Honavar S, Selva D. Orbital tuberculosis: A review of the literature. Orbit 2008;27:267-77.
5.	Singh M, Gupta N, Zadeng Z, Singh N, Kaur M, Gupta P. Long-term outcomes of oral anti-tubercular therapy in patient with tubercular dacryoadenitis: A case series. Ocul Immunol Inflamm 2019;27:1016-22.
6.	Toledano N, Stoica BTL, Saavedra IG, Saenz SG, Conde E. Tuberculous dacryoadenitis unveils HIV infection. Can J Ophthalmol 2013;48:e128-30.
7.	Bist SS, Luthra M, Agarwal V, Bharti B. Isolated lacrimal gland tuberculosis: A rare clinical entity in a young child. Online J Health Allied Scs 2016;15:12.
8.	Hina K, Seema M, Taskin K, Mehvish H. Acute lacrimal gland swelling with intracranial extension and without any neurological features – A rare case report. Saudi J Ophthalmol 2018;32:253-6.
9.	Powers HR, Diaz MA, Mendez JC. Tuberculosis presenting as dacryoadenitis in the USA. BMJ Case Rep 2019;12:e231694.
10.	Ayari R, Amri R, Chalbi E, Sbai MA. Bilateral tuberculous dacryoadenitis. Int J Mycobacteriol 2020;9:332-4. [PUBMED] [Full text]
11.	Güzel MZ, Kilic A, Arslan H. Palatal fistula: A complication of primary tuberculous dacryoadenitis. J Plast Reconstr Aesthet Surg 2008;61:1105-7.
12.	Mortada A. Tuberculoma of the orbit and lacrimal gland. Br J Ophthalmol 1971; 55:565-7.
13.	Baghdassarian SA, Zakharia H, Asdourian KK. Report of a case of bilateral caseous tuberculous dacryoadenitis. Am J Ophthalmol 1972;74:744-6.
14.	Sen DK. Tuberculosis of the orbit and lacrimal gland: A clinical study of 14 cases. J Pediatr Ophthamol Strabismus 1980;17:232-8.
15.	Panda A, Singhal V. Tuberculosis of lacrimal gland. Ind J Pediatr 1989; 56:531-3.
16.	Mohammad AE, Al-Hussaini MK, Karoosh SS, El-Moneim MT, Carey BJ, Karcioglu ZA. Tuberculosis of the orbit and lacrimal gland. Orbit 1992;11:199-204.
17.	van Assen S, Lutterman JA. Tuberculous dacryoadenitis: A rare manifestation of tuberculosis. Netherlands J Med 2002;60:327–9.
18.	Schmoll C, Macrae M, Mulvihill A, Murray R, Cunningham S, McKenzie K. Tuberculous dacryoadenitis in a Scottish teenager. Br J Ophthalmol 2009;93:137-8.
19.	Chakraborty AK.Epidemiology of tuberculosis: Current status in India Indian. J Med Res 2004;120:248-76.
20.	Babu K, Mukhopadhyay M, Bhat SS, Chinmayee J. Orbital and adnexal tuberculosis: A case series from a South Indian population. J Ophthalmic Inflamm Infect 2014;22:12.
21.	Ruman-Colombier M, Crisinel PA, Cohen-Dumani N, Ceschi G, Rochat Guignard I. Bilateral dacryoadenitis: don't forget tuberculosis! Pediatr Infect Dis J 2017;36:117-9.