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Year 2020

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Desai and Shah: Pleomorphic adenoma in lacrimal gland: Histopathological study


Introduction

The lacrimal gland is non-capsulated, multilobulated secretory gland situated in the lacrimal gland fossa. Lesion and tumors of lacrimal gland occupy almost 18% of all orbital tumor.1 Lacrimal gland tumor are relatively rare so most publish article describe single case or small series, But in our study we studied almost 38 cases in duration of more than 10 years. Structure of lacrimal gland is similar to salivary gland so for the classification of the lacrimal gland tumor is done according to the WHO (world health organisation) classification of salivary gland tumors.2

Who classification of salivary gland tumors

Malignant epithelial tumors

  1. Acinic cell carcinoma

  2. Mucoepidermoid carcinoma

  3. Adenoid cystic carcinoma

  4. Polymorphous low grade adenocarcinoma

  5. Epithelial-myoepithelial carcinoma

  6. Clear cell carcinoma, not otherwise specific

  7. Basal cell adenocarcinoma

  8. Sebaceous carcinoma

  9. Sebaceous lymphadenocarcinoma

  10. Cystadenocarcinoma

  11. Low grade cribriform cystadenocarcinoma

  12. Mucinous adenocarcinoma

  13. Oncocytic carcinoma

  14. Salivary duct carcinoma

  15. Adenocarcinoma, not otherwise specified

  16. Myoepithelial carcinoma

  17. Carcinoma ex pleomorphic adenoma

  18. Carcinosarcoma

  19. Metastasizing pleomorphic adenoma

  20. Squamous cell carcinoma

  21. Small cell carcinoma

  22. Large cell carcinoma

  23. Lymphoepithelial carcinoma

  24. Sialoblastoma

Benign epithelial tumors

  1. Pleomorphic adenoma

  2. Myoepithelioma

  3. Basal cell adenoma

  4. Warthin”s tumor

  5. Basal cell adenoma

  6. Oncocytoma

  7. Canalicular adenoma

  8. Sebaceous adenoma

  9. Ductal papilloma

  10. Inverted ductal papilloma

  11. Intraductal papilloma

  12. Sialadenoma papilliferum

  13. Cyst adenoma

  14. Lymph adenoma

(a) Sebaceous (b) Non-sebaceous

Secondary tumors

Pleomorphic adenoma of lacrimal gland is a well define benign tumour characterised by a mixed appearance of proliferated epithelial ducts and myxoid, mucoid or cartilaginous area containing myoepithelial cells. Tumour arise from progenitor ductal cell capable of both epithelial and myoepithelial differentiation.1 Pleomorphic adenomas the most common primary epithelial tumor of lacrimal gland and arise most commonly from orbital lobe (90%), and occasionally from palpebral lobe and rarely from accessory lacrimal gland.3,4,5 Most patient present with slowly progressive, painless mass at the supratemporal orbit. Grossly pleomorphic adenoma are pseudo capsulated, smooth surfaced tumours and cut surface shows alternating mucinous and fibrous areas with occasional cystic structure. A layer of compressed connective tissue surrounds the tumor and focal small tumor projections known as “BOSSELATIONS”. Microscopically characteristic features are the presence of epithelium – lined duct like structure and outer layer of darker myoepithelial cells. Mucoid, basophilic material accumulate among the myoepithelial cells and between the epithelial ducts like structure. In some cases foci of chondroid like material and even calcification are observed. The epithelial cell may form solid cords and even show squamous differentiations and cyst formations. The microscopic diagnosis of Pleomorphic adenoma is easy but sometimes confuse with myoepithelial adenoma and oncocytic adenomas. Pleomorphic Adenoma are DNA diploid tumor with low expression of the p53 gene.6 P21 Expression is apparently related to progression of the pleomorphic adenoma of the lacrimal gland.one study showed recurrent chromosomal abnormalities involving chromosome 3, 8, 9, and 12 similar to those found in salivary gland tumours.7,8 Sensitivity of preoperative diagnosis is approximately 90%. The overall 15 year recurrence rate is less than 3% if the tumor removed at the initial surgery. Despite the presence of recurrence the survival rate at 15 years is almost 100%.

Materials and Methods

A retrospective study of 38 cases of pleomorphic adenoma of lacrimal gland tumours were studied over a period of 10 years at tertiary care institute. A detailed clinical history was obtained with specific focus on age, sex and histopathological examination include macroscopic examination and microscopic examination with special stains like PAS (periodic acid Schiff ’s) stain and immunohistochemistry where it required. For macroscopic examination specimen were fixed in 10% buffered formalin and then processed, sectioned and stained with H & E (haematoxylin and eosin stain) stain. For Microscopic examination H & E stained sections were studied from different areas. Sections were studied for diagnosis of tumor, others histological features and differention and for individual tumor cells type which includes size, shape, characters of nucleus, nucleolus.

Result

Total 57 cases of lacrimal gland tumor were studied among them pleomorphic adenoma consist of 38 cases (66.66%) cases. Other tumor were diagnosed are adenocystic carcinoma, mucoepidermoid carcinoma, squamous cell carcinoma but in this study our focus on pleomorphic adenoma.so prevalence of pleomorphic adenoma is 66.66%. Present study consists of 38 cases of pleomorphic adenoma of lacrimal gland. When studied according to the age group, then 0-20 years groups consist of 2 cases (5.3%), 21-40 years age group consist of 18 cases (47.3%), 41-60 years age groups consist of 13 cases (34.2%) cases, and > 60 years age group consist of 5 cases (13.2%). When 38 cases studied according to sex then out of 38 cases, 26 cases (68.4%) were in female and 12 cases (31.6%) were in males. On gross examination Pleomorphic adenoma typically have a pseudo capsule surrounding the mass lesion. Histologically, pleomorphic adenoma shows varying proportions of epithelial and mesenchymal components. The epithelial cells form characteristic double layer ducts, acini, strands, irregular tubules and surrounding myoepithelial cells with background of loose myxoid tissue containing chondroid and rarely bone formation. When divided accordingly histological features then Myxoid differentiation in 33(94%)cases, Squamous metaplasia in 18 cases (47%), Chondroid differentiation in 16 cases(42%), Cystic degeneration in 12 cases (33%), Calcification in 3 cases (8%), Cellular atypia in 2 cases(6%), Osseous metaplasia in 1 cases (2.6%), Hyline changes in 17 cases (45%), Predominantly cellular appearance in 21 cases (55.2%), Predominantly stromal appearance in 1 cases (2.6%), Mixed (cellular + stromal) (3) 3 cases (7.9%).

Table 1
Age group (years) Number Percentage (%)
0-20 2 5.3%
21-40 18 47.3%
41-60 13 34.2%
>60 5 13.2%
Total 38 100%

Age distribution in pleomorphic adenoma in lacrimal gland

Table 2
Age group (years) No of cases in Male No of cases in Female Total no case
0-20 0 02 02
21-40 6 12 18
41-60 4 09 13
>60 2 03 05
Total 12 (31.6%) 26 (68.4%) 38

Sex and age distribution in pleomorphic adenoma of lacrimal gland

Table 3
Myxoid differentiation 33 94%
Squamous metaplasia 18 47%
Chondroid differentiation 16 42%
Cystic degeneration 12 33%
Calcification 3 8%
Cellular atypia 2 6%
Osseous metaplasia 1 2.63%
Hyline changes 17 45%
Predominantly cellular 21 55.26%
Predominantly stromal 1 2.63%
Mixed (cellular + stromal) 3 7.9%

Histological features presentation in pleomorphic adenoma of lacrimal gland

Figure 1

Myxoid area in pleomorphic adenoma (Myxoid Differantation, H & E STAIN, 40 X)

https://s3-us-west-2.amazonaws.com/typeset-media-server/0ed0a008-2b45-4e5a-8834-618110a3afd0image1.jpeg

Figure 2

Chondroid differentiation in pleomorphic adenoma (CHONDROID DIFF, H & E STAIN, 40 X)

https://s3-us-west-2.amazonaws.com/typeset-media-server/0ed0a008-2b45-4e5a-8834-618110a3afd0image2.jpeg

Figure 3

Microscopic appearance of pleomorphic adenoma (Mixed & stromal component, H & E Stain, 40 X)

https://typeset-prod-media-server.s3.amazonaws.com/article_uploads/d31e8ef9-5127-4014-bdc8-7edff49cf151/image/63eb00fb-58b2-42bb-b779-50d41d89e62b-u50.png

Figure 4

Squamous differentiation in p leomorphic adenoma (SQUAMOUS DIFF, H & E STAIN, 40 X)

https://s3-us-west-2.amazonaws.com/typeset-media-server/0ed0a008-2b45-4e5a-8834-618110a3afd0image4.jpeg

Discussion

A study of 38 cases of pleomorphic adenoma of lacrimal gland over 10 years was done. when cases are divided according to age and sex then results are summarised in Table 2. So among total 38 cases the maximum cases in age group is 21-40 years age group (Table 1) contain 18 cases (47.3%) as some study9,10 suggest that pleomorphic adenoma is most common in middle age, second age group which contain most cases is 41-60 years age group contain 13 cases (34.2%), and after that > 60 years age group contain 5 cases (13.2%), and followed by 0-20 years contain 2 cases (5.3%). So pleomorphic adenoma is least common in early age in childhood in our study. Secondly when focus in sex distribution of tumor in specific age group as well as in total cases result is out of 38 cases total 26 cases (68.4%) are seen in female and 12 cases (31.6%) are seen in male. And when divided into specific age group and then incidence noted according to sex then also individual age group shows clear cut female predominance Table 2. Now on when focused on various histopathological features on microscopy study results are following. On microscopy 33 cases (94%) cases shows myxoid differentiation, 16 cases(42%) shows chondroid differentiation, 17 cases (45%) shows hyaline changes, 18 cases(47%) shows squamous metaplasia, Figure 4 3 cases (8%) shows calcification, 2 cases (6%) shows cellular atypia, 12 cases (33%) shows cystic degenerative changes, 1 case(2.6%) shows osseous metaplasia Table 3. Other histological changes are also detected but they are not in notifiable amount. Among 38 cases 21 cases are predominantly cellular component are seen, when in 1 cases predominantly stromal component are seen, and in 3 cases mixed pattern are seen. So on above result the most common histological change is noticed is the myxoid differentiation (94%), second most common squamous metaplasia in 47% cases, followed by hyaline change (45%), and chondroid differentiation (42%). So for the summary the pleomorphic adenoma is relatively common in female and common presenting age group is middle age and most common histological feature present is the myxoid differentiation.

Conclusion

A retrospective study of 38 cases of pleomorphic adenoma of lacrimal gland shows pleomorphic adenoma is the most common benign tumor of the lacrimal gland as well as most common tumor of the lacrimal gland. The peak age for pleomorphic adenoma is the middle age mostly 21-40 year age groups contain 47.3% cases, if bar is raised and group is made 21-60 years middle age groups then more than 80% or exactly 81.5% cases fall in that group and extreme of age means in childhood and old age this tumour is less prevalent but incidence are there. And after that focus on gender we found that out of 38 cases 26 cases are seen in female means 68.4% cases are seen in female show it indicate slight female preponderance not even in total cases but in individual age group case also shows female preponderances so pleomorphic adenoma is slight more prevalent in to the Female. On comment in histological features on microscopy shows most common histological features noted was myxoid differentiation which is seen in 94% cases, followed by the squamous metaplasia in 47% cases, other histological features are in less significant amount.

Source of funding

None.

Conflict of interest

None.

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