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A closer look at heterotopic sites of pleomorphic adenoma, particularly in women: fide, sed cui vide?

The majority of the minor salivary gland tumors are malignant but they are not frequent in overall salivary gland tumors. We read with a great deal the article entitled “Heterotopic sites of pleomorphic adenoma.” This beneficial report on two vignette cases of women determined heterotopic sites and exhibited two locations, i.e., the level IIA of the second compartment of the neck and the lower lip. Kaur and colleagues declared in their recent work that the provisional diagnosis for a 46-year-old woman with lower lip swelling performed by the clinician was a mucocele, a priori. Afterward, the authors stated that fine-needle aspiration (FNA) cytology was applied, and a diagnosis of pleomorphic adenoma was inferred. Finally, they reported that the patient was lost to follow-up, a posteriori11. Kaur J, Kaur M. Heterotopic sites of pleomorphic adenoma. J Family Med Prim Care. 2023;12(5):1009-11. https://doi.org/10.4103/jfmpc.jfmpc_1591_22
https://doi.org/10.4103/jfmpc.jfmpc_1591...
. Kaur et al. also emphasized that pleomorphic adenoma, per se, is rarely seen in males; the lip is a rare site of occurrence for this phenomenon; the lower lip is a more seldom location than the upper one; the upper lip tumors are more commonly benign, whereas the lower lip ones are more frequently malignant due to their difference in embryonic development; and the complete wide local resection of the tumor with negative margins is the treatment of choice11. Kaur J, Kaur M. Heterotopic sites of pleomorphic adenoma. J Family Med Prim Care. 2023;12(5):1009-11. https://doi.org/10.4103/jfmpc.jfmpc_1591_22
https://doi.org/10.4103/jfmpc.jfmpc_1591...

2. Torske K. Benign neoplasms of the salivary glands. In: Thompson LDR, Goldblum JR, editors. Head and neck pathology (foundations of diagnostic pathology). 1st ed. Philadelphia (PA): Elsevier’s Health Sciences, Churchill Livingstone; 2006. p. 295-320.

3. Sengul I, Sengul D, Aribas D. Pleomorphic adenoma of the lower lip: a rare site of location. N Am J Med Sci. 2011;3(6):299-301. https://doi.org/10.4297/najms.2011.3299
https://doi.org/10.4297/najms.2011.3299...
-44. Sengul I, Sengul D. Pleomorphic adenoma of the lower lip: a review. N Am J Med Sci. 2011;3(12):536-9. https://doi.org/10.4297/najms.2011.3536
https://doi.org/10.4297/najms.2011.3536...
. They remarked in their work that they opted for an FNA procedure and lost her to follow-up as she possessed a benign cytology. Nevertheless, age over 40 years, male sex, mass over 20 mm in diameter, locations of the deep lobe, and inefficient first surgical intervention might be the influencing factors of multiple recurrences in pleomorphic adenoma11. Kaur J, Kaur M. Heterotopic sites of pleomorphic adenoma. J Family Med Prim Care. 2023;12(5):1009-11. https://doi.org/10.4103/jfmpc.jfmpc_1591_22
https://doi.org/10.4103/jfmpc.jfmpc_1591...

2. Torske K. Benign neoplasms of the salivary glands. In: Thompson LDR, Goldblum JR, editors. Head and neck pathology (foundations of diagnostic pathology). 1st ed. Philadelphia (PA): Elsevier’s Health Sciences, Churchill Livingstone; 2006. p. 295-320.

3. Sengul I, Sengul D, Aribas D. Pleomorphic adenoma of the lower lip: a rare site of location. N Am J Med Sci. 2011;3(6):299-301. https://doi.org/10.4297/najms.2011.3299
https://doi.org/10.4297/najms.2011.3299...

4. Sengul I, Sengul D. Pleomorphic adenoma of the lower lip: a review. N Am J Med Sci. 2011;3(12):536-9. https://doi.org/10.4297/najms.2011.3536
https://doi.org/10.4297/najms.2011.3536...
-55. Krolls SO, Boyers RC. Mixed tumors of salivary glands. Long-term follow-up. Cancer. 1972;30(1):276-81. https://doi.org/10.1002/1097-0142(197207)30:1<276::aid-cncr2820300138>3.0.co;2-v
https://doi.org/10.1002/1097-0142(197207...
. In addition, possible malignant transformation is estimated at the rate of 2–7%. Of note, deficiency of encapsulation might lead to a rupture and seeding of the mentioned tumoral structure22. Torske K. Benign neoplasms of the salivary glands. In: Thompson LDR, Goldblum JR, editors. Head and neck pathology (foundations of diagnostic pathology). 1st ed. Philadelphia (PA): Elsevier’s Health Sciences, Churchill Livingstone; 2006. p. 295-320.

3. Sengul I, Sengul D, Aribas D. Pleomorphic adenoma of the lower lip: a rare site of location. N Am J Med Sci. 2011;3(6):299-301. https://doi.org/10.4297/najms.2011.3299
https://doi.org/10.4297/najms.2011.3299...
-44. Sengul I, Sengul D. Pleomorphic adenoma of the lower lip: a review. N Am J Med Sci. 2011;3(12):536-9. https://doi.org/10.4297/najms.2011.3536
https://doi.org/10.4297/najms.2011.3536...
. Herewith, we postulate that at least a meticulous and long-term follow-up is crucial for not overlooking any recurrence or malignant transformation, particularly on the topographic localization at the lower lip, by choosing a non-surgical approach, which might bring blurred lines66. Sengul I, Sengul D. Blurred lines for management of thyroid nodules in the era of atypia of undetermined significance/ follicular lesion of undetermined significance: novel subdivisions of categories IIIA and IIIB in a possible forthcoming The Bethesda System for Reporting Thyroid Cytopathology, 3rd edition; amending versus unnecessary? Rev Assoc Med Bras (1992). 2021;67(10):1385-6. https://doi.org/10.1590/1806-9282.20210763
https://doi.org/10.1590/1806-9282.202107...
in the minds of providers. As a matter of fact, this issue merits further investigation. We thank Kaur et al.11. Kaur J, Kaur M. Heterotopic sites of pleomorphic adenoma. J Family Med Prim Care. 2023;12(5):1009-11. https://doi.org/10.4103/jfmpc.jfmpc_1591_22
https://doi.org/10.4103/jfmpc.jfmpc_1591...
for their valuable study.

ACKNOWLEDGMENTS

We thank all of the study participants.

REFERENCES

  • 1.
    Kaur J, Kaur M. Heterotopic sites of pleomorphic adenoma. J Family Med Prim Care. 2023;12(5):1009-11. https://doi.org/10.4103/jfmpc.jfmpc_1591_22
    » https://doi.org/10.4103/jfmpc.jfmpc_1591_22
  • 2.
    Torske K. Benign neoplasms of the salivary glands. In: Thompson LDR, Goldblum JR, editors. Head and neck pathology (foundations of diagnostic pathology). 1st ed. Philadelphia (PA): Elsevier’s Health Sciences, Churchill Livingstone; 2006. p. 295-320.
  • 3.
    Sengul I, Sengul D, Aribas D. Pleomorphic adenoma of the lower lip: a rare site of location. N Am J Med Sci. 2011;3(6):299-301. https://doi.org/10.4297/najms.2011.3299
    » https://doi.org/10.4297/najms.2011.3299
  • 4.
    Sengul I, Sengul D. Pleomorphic adenoma of the lower lip: a review. N Am J Med Sci. 2011;3(12):536-9. https://doi.org/10.4297/najms.2011.3536
    » https://doi.org/10.4297/najms.2011.3536
  • 5.
    Krolls SO, Boyers RC. Mixed tumors of salivary glands. Long-term follow-up. Cancer. 1972;30(1):276-81. https://doi.org/10.1002/1097-0142(197207)30:1<276::aid-cncr2820300138>3.0.co;2-v
    » https://doi.org/10.1002/1097-0142(197207)30:1<276::aid-cncr2820300138>3.0.co;2-v
  • 6.
    Sengul I, Sengul D. Blurred lines for management of thyroid nodules in the era of atypia of undetermined significance/ follicular lesion of undetermined significance: novel subdivisions of categories IIIA and IIIB in a possible forthcoming The Bethesda System for Reporting Thyroid Cytopathology, 3rd edition; amending versus unnecessary? Rev Assoc Med Bras (1992). 2021;67(10):1385-6. https://doi.org/10.1590/1806-9282.20210763
    » https://doi.org/10.1590/1806-9282.20210763
  • Funding: none.

Publication Dates

  • Publication in this collection
    04 Mar 2024
  • Date of issue
    2024

History

  • Received
    05 Sept 2023
  • Accepted
    24 Sept 2023
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