Mucoepidermoid Carcinoma in Warthin Tumor of the Parotis in Childhood: A Case Report and Review of the Literature

Çıtak, Elvan Çağlar; Yılmaz, Erdem; Sağcan, Fatih; Bozkurt, Funda; Arpacı, Rabia Bozdoğan; Vayısoğlu, Yusuf

doi:10.1097/mph.0000000000001303

Cited by 11 publications

(15 citation statements)

References 18 publications

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“…Clinically, our data showed that with Warthin-like MEC, there is a slight female preponderance (male to female ratio, 4:5), whereas in other reports on Warthin-like MEC, there was a strong female preponderance. 20,21,23,24,29 For patients with nonsebaceous LAD, the entity was more common in males, which is also different from other reports. 6 In our study, the patients with Warthin-like MEC had a wider age distribution (10-75 years) than the patients with nonsebaceous LAD (47-68 years).…”

Section: Discussionmentioning

confidence: 68%

Reevaluation of Salivary Lymphadenoma: A Subgroup Identified as Warthin-like Mucoepidermoid Carcinoma Following Molecular Investigation for MAML2 Rearrangement

Zhang

et al. 2020

Archives of Pathology &Amp; Laboratory Medicine

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Context.— Both salivary lymphadenomas (LADs) and Warthin-like mucoepidermoid carcinoma (MEC) contain components of epithelium and lymphoid stroma and their differential diagnosis can be difficult on the basis of morphology alone. Objective.— To clarify whether Warthin-like MEC was diagnosed as a LAD, and to compare their clinicopathologic features. Design.— A total of 16 LAD cases were analyzed for MAML2 rearrangement by using fluorescence in situ hybridization, and the clinical, histologic, immunohistochemical, and prognostic features were compared between MAML2 rearrangement–positive and MAML2 rearrangement–negative groups. Results.— Among the 16 cases investigated, 9 harbored a MAML2 rearrangement and were reclassified as Warthin-like MEC. The remaining 7 cases were classified as LADs with 1 nonsebaceous and 6 sebaceous cases. The patients with Warthin-like MEC had a wider age range (10–75 years) than the patients with LADs (36–68 years). Histologically, 2 of the 9 Warthin-like MECs (22.2%) showed focal invasion, whereas all the LADs had complete capsules. Warthin-like MECs exhibited a diverse epithelial cell morphology, including basaloid, glandular, cuboidal, epidermoid, with mucinous cells, although these cytologic features were seen only focally in some cases. Nonsebaceous LAD was composed of basaloid and glandular epithelial cells predominantly. In sebaceous LAD, the epithelial cells were composed of basaloid and large foamy sebaceous cells. In all cases, the stroma was composed mainly of lymphocytes accompanied by lymphoid follicles, although plasma cell infiltration could be much heavier in Warthin-like MEC. All the patients had a good outcome after a longer follow-up (3∼166 months). Conclusions.— Warthin-like MEC can be misdiagnosed as a LAD owing to overlap in clinicopathologic features of the 2 entities. Careful histologic evaluation and detection of MAML2 rearrangement can facilitate their differential diagnosis.

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Section: Discussionmentioning

confidence: 68%

Reevaluation of Salivary Lymphadenoma: A Subgroup Identified as Warthin-like Mucoepidermoid Carcinoma Following Molecular Investigation for MAML2 Rearrangement

Zhang

et al. 2020

Archives of Pathology &Amp; Laboratory Medicine

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“…The tumor has been reported to transform to epithelial and lymphoid malignancies in extreme rare cases. [ 15 16 17 ]…”

Section: Discussionmentioning

confidence: 99%

Incipient Warthin tumor of intraparotid lymph node in a patient with squamous cell carcinoma of the external auditory canal: An incidental finding

Pandiar

Aravind

Nayanar

et al. 2020

Natl J Maxillofac Surg

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The occurrence of intranodal salivary gland neoplasm is uncommon; squamous cell carcinoma (SCC) of the external auditory canal (EAC) is another rare occurrence. Clinically, SCC of EAC presents with symptoms similar to other benign otologic conditions. A case of Stage I SCC in EAC region is presented here in a 60-year-old male patient with incidental intranodal Warthin tumor along with the histological differential diagnosis. The patient is being followed up. There is no evidence of recurrence 1 year and 11 months after surgery.

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“…The main differential of such types of lesions is the malignant transformation of WT, metaplastic changes in WT and the Warthin-like variant of MEC. The coexistence of WT and MEC is rare, with only 29 cases reported worldwide 4 - 6. Malignant transformation of the epithelial component of WT, such as squamous cell carcinoma, MEC, adenocarcinoma, and undifferentiated, poorly differentiated, and anaplastic carcinoma is extremely rare and only seen in 0.3% of cases 4.…”

Section: Discussionmentioning

confidence: 99%

“…Cytogenetic studies of the tumor showing both WT and MEC characteristics identified specific chromosomal translocations: t(11;19) (q21;p13), and t(11;15)(q21;q26) result in novel fusion oncogenes CRTC1-MAML2 and CRTC3-MAML2 , respectively. These fusion genes act as transcription factors on the Notch and CREB regulatory pathway 6. Ishibashi et al2 reported 15 cases of WT with diffuse squamous metaplasia, 5 of which were reclassified into low-grade MEC after testing positive for CRTC1-MAML2 gene fusion.…”

Section: Discussionmentioning

confidence: 99%

“…MEC grading is based on the percentage of mucous cells into the low, intermediate, and high grade. Low-grade lesions are usually managed with surgery alone while high-grade lesions or high-risk lesions (with neural invasion, necrosis, mitotic figures, anaplasia), and margin-positive lesions require adjuvant radiotherapy 6. No case of metastases has been reported in patients with MEC coexisting with WT of the parotid gland treated with margin-free excision.…”

Section: Discussionmentioning

confidence: 99%

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Warthin-like mucoepidermoid carcinoma of the parotid gland: a diagnostic and therapeutic dilemma

Balasubiramaniyan¹,

Sultania²,

Sable³

et al. 2019

Autops. Case Rep.

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Mucoepidermoid carcinoma (MEC) is the most common malignant salivary gland tumor. Although the parotid gland is the most common site of involvement, other major salivary glands and the minor salivary glands—most commonly of the palate—also can be involved. The management of mucoepidermoid carcinoma depends on the grade of the tumor and the adequacy of resection. We present the case of a 56-year-old female presenting a painless progressive cheek mass over 2 months. Imaging and fine-needle aspiration cytology provided the diagnosis of Mucoepidermoid carcinoma. A superficial parotidectomy was done, and the histopathology revealed a predominantly cystic tumor with a bilayered epithelium of oncocytic and basal cells. Moderate nuclear pleomorphism with infiltration of atypical squamous cells in few glandular cysts was seen. Special staining revealed the presence of intracellular mucin. A diagnosis of Warthin-like variant of MEC was made, based on these findings. After the surgical procedures, the patient is disease-free at 8 months of follow-up. The Warthin-like variant is a rare variant of MEC with fewer than 10 cases described in the English literature. Various differential diagnoses include the malignant transformation of Wartin tumor (WT), squamous metaplasia of WT, and metastasis from a distant primary. We emphasize the role of routine microscopy in identifying rare variants of common malignancies. Even though translocation studies are helpful in diagnosis, the typical histopathological findings should confirm it.

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Mucoepidermoid Carcinoma in Warthin Tumor of the Parotis in Childhood: A Case Report and Review of the Literature

Cited by 11 publications

References 18 publications

Reevaluation of Salivary Lymphadenoma: A Subgroup Identified as Warthin-like Mucoepidermoid Carcinoma Following Molecular Investigation for MAML2 Rearrangement

Reevaluation of Salivary Lymphadenoma: A Subgroup Identified as Warthin-like Mucoepidermoid Carcinoma Following Molecular Investigation for MAML2 Rearrangement

Incipient Warthin tumor of intraparotid lymph node in a patient with squamous cell carcinoma of the external auditory canal: An incidental finding

Warthin-like mucoepidermoid carcinoma of the parotid gland: a diagnostic and therapeutic dilemma

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