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Clinical research on benign lymphoepithelial lesions of lacrimal gland in 20 Chinese patients.

Ma JM, Li J, Ge X, Li M, Li JR, Wang XN - Chin. Med. J. (2015)

Bottom Line: Most of the patients involved bilateral lacrimal glands.High serum IgG4 level and abundant IgG4-positive plasma cell infiltration are the important features, which can be found in most of BLEL patients.Surgery combined with glucocorticoids is an efficient treatment strategy.

View Article: PubMed Central - PubMed

Affiliation: Beijing Tongren Eye Center, Beijing Tongren Hospital, Capital Medical University, Beijing Ophthalmology and Visual Sciences Key Laboratory, Beijing 100730, China.

ABSTRACT

Background: Benign lymphoepithelial lesion (BLEL) is characterized by symmetric bilateral swelling of the lacrimal and salivary glands and considered a subtype of immunoglobulin G4 (IgG4)-related sclerosing disease, the etiology and pathogenesis of which has not been determined. The purpose of the present study was to analyze the clinical features of BLEL in the lacrimal gland and the relationship between the serum level of IgG4 and BLEL.

Methods: Twenty consecutive patients with BLEL diagnosed in Department of Ophthalmology at Beijing Tongren Hospital, Capital Medical University between January 2012 and December 2013 were observed. The clinical features, imaging findings, laboratory tests, treatments and follow-up status of these 20 consecutive patients were analyzed.

Results: In all 20 patients, the ratio of male to female was 1:4, the ages ranged from 28 to 57 years, the ratio of unilateral to bilateral eyes involvement was 1:4, and painless uncongested symmetrical swelling of the upper eyelid was the main clinical manifestation. Orbital magnetic resonance imaging (MRI) showed that all patients involved lacrimal gland, which were obviously enlarged with equal signals in T1W images and T2W images and obvious enhancement on contrast MRI. Extraocular muscles were involved in 5 patients, salivary gland in 8 patients, and frontal nerve in 3 patients. Serum IgG4 concentration was elevated in 18 patients. The treatment strategy mainly included surgery and steroid administration. Three patients were lost to follow-up, 17 patients reached complete response, and no recurrence was observed.

Conclusions: Eyelid swelling is the typical symptom of BLEL. Most of the patients involved bilateral lacrimal glands. High serum IgG4 level and abundant IgG4-positive plasma cell infiltration are the important features, which can be found in most of BLEL patients. Surgery combined with glucocorticoids is an efficient treatment strategy.

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Related in: MedlinePlus

Histopathology of benign lymphoepithelial lesion (patient 2). (a) Marked lymphoplasmacytic infiltration with lymphoid follicles and the destruction and atrophy of the lacrimal gland lobules with periductal fibrosis were observed (hematoxylin-eosin staining, original magnification ×40). (b) Immunostaining for immunoglobulin G4 showed abundant IgG4-positive plasma cells infiltrated the lesion (original magnification ×20).
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Figure 3: Histopathology of benign lymphoepithelial lesion (patient 2). (a) Marked lymphoplasmacytic infiltration with lymphoid follicles and the destruction and atrophy of the lacrimal gland lobules with periductal fibrosis were observed (hematoxylin-eosin staining, original magnification ×40). (b) Immunostaining for immunoglobulin G4 showed abundant IgG4-positive plasma cells infiltrated the lesion (original magnification ×20).

Mentions: Prominent lymphoplasmacytic infiltration with lymphoid follicles was observed with the destruction and atrophy of the lacrimal gland lobules and periductal fibrosis [Figure 3a]. Immunostaining for IgG4 expression showed that numerous IgG4-positive plasma cells infiltrated the lesion [Figure 3b]. There were 16 patients of moderate (80%), 2 patients of marked, 1 patient of mild, and 1 patient of negative infiltration; and the degree of IgG4 expression in both tissue and blood was accordant [Table 3].


Clinical research on benign lymphoepithelial lesions of lacrimal gland in 20 Chinese patients.

Ma JM, Li J, Ge X, Li M, Li JR, Wang XN - Chin. Med. J. (2015)

Histopathology of benign lymphoepithelial lesion (patient 2). (a) Marked lymphoplasmacytic infiltration with lymphoid follicles and the destruction and atrophy of the lacrimal gland lobules with periductal fibrosis were observed (hematoxylin-eosin staining, original magnification ×40). (b) Immunostaining for immunoglobulin G4 showed abundant IgG4-positive plasma cells infiltrated the lesion (original magnification ×20).
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4836253&req=5

Figure 3: Histopathology of benign lymphoepithelial lesion (patient 2). (a) Marked lymphoplasmacytic infiltration with lymphoid follicles and the destruction and atrophy of the lacrimal gland lobules with periductal fibrosis were observed (hematoxylin-eosin staining, original magnification ×40). (b) Immunostaining for immunoglobulin G4 showed abundant IgG4-positive plasma cells infiltrated the lesion (original magnification ×20).
Mentions: Prominent lymphoplasmacytic infiltration with lymphoid follicles was observed with the destruction and atrophy of the lacrimal gland lobules and periductal fibrosis [Figure 3a]. Immunostaining for IgG4 expression showed that numerous IgG4-positive plasma cells infiltrated the lesion [Figure 3b]. There were 16 patients of moderate (80%), 2 patients of marked, 1 patient of mild, and 1 patient of negative infiltration; and the degree of IgG4 expression in both tissue and blood was accordant [Table 3].

Bottom Line: Most of the patients involved bilateral lacrimal glands.High serum IgG4 level and abundant IgG4-positive plasma cell infiltration are the important features, which can be found in most of BLEL patients.Surgery combined with glucocorticoids is an efficient treatment strategy.

View Article: PubMed Central - PubMed

Affiliation: Beijing Tongren Eye Center, Beijing Tongren Hospital, Capital Medical University, Beijing Ophthalmology and Visual Sciences Key Laboratory, Beijing 100730, China.

ABSTRACT

Background: Benign lymphoepithelial lesion (BLEL) is characterized by symmetric bilateral swelling of the lacrimal and salivary glands and considered a subtype of immunoglobulin G4 (IgG4)-related sclerosing disease, the etiology and pathogenesis of which has not been determined. The purpose of the present study was to analyze the clinical features of BLEL in the lacrimal gland and the relationship between the serum level of IgG4 and BLEL.

Methods: Twenty consecutive patients with BLEL diagnosed in Department of Ophthalmology at Beijing Tongren Hospital, Capital Medical University between January 2012 and December 2013 were observed. The clinical features, imaging findings, laboratory tests, treatments and follow-up status of these 20 consecutive patients were analyzed.

Results: In all 20 patients, the ratio of male to female was 1:4, the ages ranged from 28 to 57 years, the ratio of unilateral to bilateral eyes involvement was 1:4, and painless uncongested symmetrical swelling of the upper eyelid was the main clinical manifestation. Orbital magnetic resonance imaging (MRI) showed that all patients involved lacrimal gland, which were obviously enlarged with equal signals in T1W images and T2W images and obvious enhancement on contrast MRI. Extraocular muscles were involved in 5 patients, salivary gland in 8 patients, and frontal nerve in 3 patients. Serum IgG4 concentration was elevated in 18 patients. The treatment strategy mainly included surgery and steroid administration. Three patients were lost to follow-up, 17 patients reached complete response, and no recurrence was observed.

Conclusions: Eyelid swelling is the typical symptom of BLEL. Most of the patients involved bilateral lacrimal glands. High serum IgG4 level and abundant IgG4-positive plasma cell infiltration are the important features, which can be found in most of BLEL patients. Surgery combined with glucocorticoids is an efficient treatment strategy.

Show MeSH
Related in: MedlinePlus