Clinical behavior of recurrent pleomorphic adenoma in the palate: a systematic review

Lopes-Santos, Gabriela; Marques, Najara Gomes de Oliveira; Tjioe, Kellen Cristine; Oliveira, Denise Tostes

doi:10.1590/acb390824

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Brasil

Acta Cirúrgica Brasileira

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Brasil

Español English

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Sumário

Review Article • Acta Bras Cir 39 • 2024 • https://doi.org/10.1590/acb390824 copy

Clinical behavior of recurrent pleomorphic adenoma in the palate: a systematic review

Authorship SCIMAGO INSTITUTIONS RANKINGS

ABSTRACT

Purpose:

This systematic review analyzed the clinical behavior and odds of malignancy of the palatal recurrent pleomorphic adenomas.

Methods:

Systematic review of patients with recurrent pleomorphic adenoma arising in the palate. Database search: MEDLINE, Scopus, Web of Science, Cochrane, EMBASE, Virtual Health Library, Google Scholar, and OpenGrey. A binomial logistic regression was performed to assess the odds of detecting recurrence five, 10 and 20 years after the treatment of primary tumor.

Results:

Thirteen studies (n = 18 patients) out of 336 were included. The recurrent pleomorphic adenoma in palate was more common in females (61.6%), average age was 49 years old (range 9–73 years old). Four patients progressed to malignant transformation. The odds ratio (OR) of detecting a recurrence at 10 (OR = 5.57; 95% confidence interval – 95%CI 1.13–27.52), and 20 years (OR = 18.78; 95%CI 3.18–110.84) after treatment of primary pleomorphic adenoma was significantly higher than at one-year follow-up.

Conclusions:

The recurrence of pleomorphic adenoma in palate remains a rare event of late occurrence. It mainly affects middle-aged female and carries a risk of malignant transformation. Although uncommon, patients with palatal pleomorphic adenoma should be warned about the possibility of recurrence or malignant transformation of tumor at advanced ages.

Key words
Neoplasms; Adenoma, Pleomorphic; Salivary Glands; Recurrence

Author, year	Gender	Primary tumor (age of patient)	Recurrent tumor (age of patient)	Time for relapse	Clinicalfeatures	Bone involvement	Treatment	Finaldiagnosis
Byars et al., 1957²³23 Byars LT, Ackerman LV, Peacock E. Tumors of salivary gland origin in children: a clinical pathologic appraisal of 24 cases. Ann Surg. 1957;146(1):40–51. https://doi.org/10.1097/00000658-195707000-00005 https://doi.org/10.1097/00000658-1957070...	Female	4 years old	9 years old	Five years	NA	NA	Surgical resection	Recurrent pleomorphic adenoma
Hellquist and Michaels, 1986²⁴24 Hellquist H, Michaels L. Malignant mixed tumour. A salivary gland tumour showing both carcinomatous and sarcomatous features. Virchows Arch A Pathol Anat Histopathol. 1986;409(1):93–103. https://doi.org/10.1007/bf00705409 https://doi.org/10.1007/bf00705409...	Female	25 years old	55 years old	30 years	Protruding and partly ulcerated with small areas of hemorrhage, measuring 20 × 15 × 15 mm	Yes	Radiotherapy and palatal resection	Chondrosarcoma
	Male	28 years old	64 years old	36 years	Mass firm and cyst-like measuring 13 × 7 × 5 mm	NA	Radiotherapy and palatal resection	Carcinoma of ductal type
Yasumoto et al., 1999²¹21 Yasumoto M, Sunaba K, Shibuya H, Kurabayashi T. Recurrent pleomorphic adenoma of the head and neck. Neuroradiology. 1999;41(4):300–4. https://doi.org/10.1007/s002340050752 https://doi.org/10.1007/s002340050752...	Male	48 years old	57 years old	Nine years	Solitary, smooth measuring approximately 40 mm	Yes	Surgical resection	Recurrent pleomorphic adenoma
	Female	46 years old	58 years old	12 years	Solitary, smooth measuring 42 mm	Yes	Surgical resection	Recurrent pleomorphic adenoma
	Female	24 years old	64 years old	40 years	Solitary, smooth measuring approximately 40 mm	Yes	Surgical resection	Recurrent pleomorphic adenoma
	Male	43 years old	66 years old	23 years	Solitary, smooth measuring 66 mm	Yes	Surgical resection	Recurrent pleomorphic adenoma
	Male	62 years old	69 years old	Seven years	Solitary, smooth measuring 66 mm	No	Surgical resection	Recurrent pleomorphic adenoma
Shaaban et al., 2001²⁵25 Shaaban H, Bruce J, Davenport PJ. Recurrent pleomorphic adenoma of the palate in a child. Br J Plast Surg. 2001;54(3):245–7. https://doi.org/10.1054/bjps.2000.3536 https://doi.org/10.1054/bjps.2000.3536...	Male	7 years old	9 years old	Two years	Non-tender, immobile and showed an ulcerated surface measuring 2 × 2 cm	No	Surgical resection	Recurrent pleomorphic adenoma
Karatzanis et al., 2005²⁶26 Karatzanis AD, Drivas EI, Giannikaki ES, Lachanas VA, Hatziioannou JK, Velegrakis GA. Malignant myoepithelioma arising from recurrent pleomorphic adenoma of the soft palate. Auris Nasus Larynx. 2005;32(4):435–7. https://doi.org/10.1016/j.anl.2005.04.001 https://doi.org/10.1016/j.anl.2005.04.00...	Female	68 years old	70 years old	Two years	Large tumor, arising from the right side of the soft palate, and blocking completely the oral cavity measuring 4.6 × 6 cm	NA	Surgical resection	Low-grade malignant myoepithelial carcinoma
Turner and Smith, 2006³³33 Turner MJA, Smith WP. An exceptionally late recurrence of pleomorphic adenoma. J Oral Maxillofac Surg. 2006;64(9):1460. https://doi.org/10.1016/j.joms.2006.05.012 https://doi.org/10.1016/j.joms.2006.05.0...	Female	19 years old	54 years old	35 years	Solitary, smooth measuring 1.2 cm	Yes	Partial maxillectomy	Recurrent pleomorphic adenoma
Negahban et al., 2006²⁷27 Negahban S, Daneshbod Y, Shishegar M. Clear cell carcinoma arising from pleomorphic adenoma of a minor salivary gland: Report of a case with fine needle aspiration, histologic and immunohistochemical findings. Acta Cytol. 2006;50(6):687–90. https://doi.org/10.1159/000326043 https://doi.org/10.1159/000326043...	Male	23 years old	53 years old	30 years	Slowly growing mass with rapid growth measuring 2.5 × 1.5 × 1 cm	NA	NA	Clear cell carcinoma
Berardi et al., 2009²⁸28 Berardi D, Scoccia A, Perfetti G, Berardi S. Recurrence of pleomorphic adenoma of the palate after sixteen years: case report and an analysis of the literature. J Biol Regul Homeost Agents. 2009;23(4):225–9.	Male	20 years old	39 years old	19 years	Swelling measuring 1.2 cm	NA	Surgical resection	Recurrent pleomorphic adenoma
Ritwik and Brannon, 2012²²22 Ritwik P, Brannon RB. A clinical analysis of nine new pediatric and adolescent cases of benign minor salivary gland neoplasms and a review of the literature. J Med Case Rep. 2012;6:287. https://doi.org/10.1186%2F1752-1947-6-287 https://doi.org/10.1186%2F1752-1947-6-28...	Female	14 years old	17 years old	Three years	Swelling measuring 1.3 cm	Yes	Surgical resection	Recurrent pleomorphic adenoma
Faisal and Mishra, 2016²⁹29 Faisal M, Mishra S. A Case of Massive Recurrent Pleomorphic Adenoma of the Palate. J Dent Oral Disord [Internet]. 2016 [cited 2023 Mar 23];2(8):1041. Available from: https://austinpublishinggroup.com/dental-disorders/fulltext/jdod-v2-id1041.php https://austinpublishinggroup.com/dental...	Female	53 years old	73 years old	20 years	Facial deformity with expansion of bilateral maxillae and palatine bones, deformed nasal bones	Yes	Surgical resection	Recurrent pleomorphic adenoma
Lima et al., 2020³⁰30 Lima FF, Bezerra CP, Rocha AC, Martins IS, Bernaola-Paredes WE. Surgical Management of Palatal Pleomorphic Adenoma (PPA) Recurrence After 10 years, Treated at a Brazilian Center - A Case Report. Ann Maxillofac Surg. 2020;10(2):533–6. https://doi.org/10.4103/ams.ams_107_20 https://doi.org/10.4103/ams.ams_107_20...	Female	36 years old	46 years old	10 years	Nodular firm measuring 2.2 × 3 cm	No	Surgical resection	Recurrent pleomorphic adenoma
Hemavathy et al., 2022³²32 Hemavathy K, Giri GVV, Subramani V, Susruthan M. Recurrent Palatal Pleomorphic Adenoma: A Case Report With a Long-Term Follow-Up. Cureus. 2022;14(6):e26363. https://doi.org/10.7759%2Fcureus.26363 https://doi.org/10.7759%2Fcureus.26363...	Female	50 years old	58 years old	7.5 years	Smooth, firm, non-tender, fixed swelling over the anterior maxilla with 3 × 2 cm of diameter	NA	Surgical resection	Recurrent pleomorphic adenoma
Nokaneng, 2022³¹31 Nokaneng EN. Invasive recurrent pleomorphic adenoma of the palate: A case report and literature review. Oral Maxillofac Surg Cases. 2022;8(4):100281. https://doi.org/10.1016/j.omsc.2022.100281 https://doi.org/10.1016/j.omsc.2022.1002...	Female	24 years old	28 years old	Four years	Asymptomatic, fluctuant swelling with 3 × 2 cm of diameter	Yes	Surgical resection	Recurrent pleomorphic adenoma

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[1] *Corresponding author: denisetostes@usp.br