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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

Clinical photograph (patient 18) showed bilateral uncongested symmetrical swelling of the upper eyelids, which was asymptomatic and nontender to palpation.
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Figure 1: Clinical photograph (patient 18) showed bilateral uncongested symmetrical swelling of the upper eyelids, which was asymptomatic and nontender to palpation.

Mentions: In all 20 patients, the male-to-female ratio was 1:4, and the ages ranged from 28 to 57 years with a median age of 45 years. The ratio of unilateral to bilateral eyes involvement was 1:4, and painless uncongested asymptomatic symmetrical swelling of the upper eyelid was the main clinical manifestation and complaint [Figure 1 and Table 1]. In addition, 3 patients had a history of salivary gland surgery, and 1 patient had a history of hepatitis C.


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)

Clinical photograph (patient 18) showed bilateral uncongested symmetrical swelling of the upper eyelids, which was asymptomatic and nontender to palpation.
© Copyright Policy - open-access
Related In: Results  -  Collection

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

Figure 1: Clinical photograph (patient 18) showed bilateral uncongested symmetrical swelling of the upper eyelids, which was asymptomatic and nontender to palpation.
Mentions: In all 20 patients, the male-to-female ratio was 1:4, and the ages ranged from 28 to 57 years with a median age of 45 years. The ratio of unilateral to bilateral eyes involvement was 1:4, and painless uncongested asymptomatic symmetrical swelling of the upper eyelid was the main clinical manifestation and complaint [Figure 1 and Table 1]. In addition, 3 patients had a history of salivary gland surgery, and 1 patient had a history of hepatitis C.

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