Publications by authors named "Ioannis G Koutlas"

66 Publications

Salivary Mucinous Adenocarcinoma Is a Histologically Diverse Single Entity With Recurrent AKT1 E17K Mutations: Clinicopathologic and Molecular Characterization With Proposal for a Unified Classification.

Am J Surg Pathol 2021 Feb 24. Epub 2021 Feb 24.

*Departments of Pathology and Oncology, The Johns Hopkins University School of Medicine, Baltimore, MD †Department of Biochemistry, Molecular Biology, and Biophysics, College of Biological Sciences ‡Howard Hughes Medical Institute, University of Minnesota §Division of Oral and Maxillofacial Pathology, School of Dentistry, University of Minnesota, Minneapolis, MN ∥Southern California Permanente Medical Group, Department of Pathology, Woodland Hills Medical Center, Woodland Hills **Departments of Orofacial Sciences, Pathology, and Radiation Oncology, University of California San Francisco, San Francisco, CA ¶Department of Pathology, University of Texas Southwestern Medical Center, Dallas, TX #Department of Pathology, Icahn School of Medicine at Mount Sinai Hospital, New York, NY.

Mucin-producing salivary adenocarcinomas were historically divided into separate colloid carcinoma, papillary cystadenocarcinoma, and signet ring cell carcinoma diagnoses based on histologic pattern, but have recently been grouped together in the adenocarcinoma not otherwise specified category. It is currently unclear if these tumors represent 1 or more distinct entities and how they are related to well-circumscribed papillary mucinous lesions with recurrent AKT1 E17K mutations that were recently described as salivary intraductal papillary mucinous neoplasm. Here, we sought to evaluate the clinicopathologic and molecular features of salivary mucinous adenocarcinomas to clarify their classification. We identified 17 invasive mucin-producing salivary adenocarcinomas, 10 with a single histologic pattern, and 7 with mixed patterns. While most tumors demonstrated papillary growth (n=15), it was frequently intermixed with colloid (n=6) and signet ring (n=3) architecture with obvious transitions between patterns. All were cytokeratin 7 positive (100%) and cytokeratin 20 negative (0%). Next-generation sequencing performed on a subset demonstrated recurrent AKT1 E17K mutations in 8 cases (100%) and TP53 alterations in 7 cases (88%). Of 12 cases with clinical follow-up (median: 17 mo), 4 developed cervical lymph node metastases, all of which had colloid or signet ring components. Overall, overlapping histologic and immunohistochemical features coupled with recurrent AKT1 E17K mutations across patterns suggests that mucin-producing salivary adenocarcinomas represent a histologically diverse single entity that is closely related to tumors described as salivary intraductal papillary mucinous neoplasm. We propose a unified mucinous adenocarcinoma category subdivided into papillary, colloid, signet ring, and mixed subtypes to facilitate better recognition and classification of these tumors.
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http://dx.doi.org/10.1097/PAS.0000000000001688DOI Listing
February 2021

Endogenous APOBEC3B overexpression characterizes HPV-positive and HPV-negative oral epithelial dysplasias and head and neck cancers.

Mod Pathol 2021 02 6;34(2):280-290. Epub 2020 Jul 6.

Department of Biochemistry, Molecular Biology and Biophysics, University of Minnesota, Minneapolis, MN, USA.

The DNA cytosine deaminase APOBEC3B (A3B) is a newly recognized endogenous source of mutations in a range of human tumors, including head/neck cancer. A3B inflicts C-to-T and C-to-G base substitutions in 5'-TCA/T trinucleotide motifs, contributes to accelerated rates of tumor development, and affects clinical outcomes in a variety of cancer types. High-risk human papillomavirus (HPV) infection causes A3B overexpression, and HPV-positive cervical and head/neck cancers are among tumor types with the highest degree of APOBEC signature mutations. A3B overexpression in HPV-positive tumor types is caused by the viral E6/E7 oncoproteins and may be an early off-to-on switch in tumorigenesis. In comparison, less is known about the molecular mechanisms responsible for A3B overexpression in HPV-negative head/neck cancers. Here, we utilize an immunohistochemical approach to determine whether A3B is turned from off-to-on or if it undergoes a more gradual transition to overexpression in HPV-negative head/neck cancers. As positive controls, almost all HPV-positive oral epithelial dysplasias and oropharyngeal cancers showed high levels of nuclear A3B staining regardless of diagnosis. As negative controls, A3B levels were low in phenotypically normal epithelium adjacent to cancer and oral epithelial hyperplasias. Interestingly, HPV-negative and low-grade oral epithelial dysplasias showed intermediate A3B levels, while high-grade oral dysplasias showed high A3B levels similar to oral squamous cell carcinomas. A3B levels were highest in grade 2 and grade 3 oral squamous cell carcinomas. In addition, a strong positive association was found between nuclear A3B and Ki67 scores suggesting a linkage to the cell cycle. Overall, these results support a model in which gradual activation of A3B expression occurs during HPV-negative tumor development and suggest that A3B overexpression may provide a marker for advanced grade oral dysplasia and cancer.
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http://dx.doi.org/10.1038/s41379-020-0617-xDOI Listing
February 2021

FET(EWSR1)-TFCP2 Rhabdomyosarcoma: An Additional Example of this Aggressive Variant with Predilection for the Gnathic Bones.

Head Neck Pathol 2021 Mar 5;15(1):374-380. Epub 2020 Jun 5.

Pediatric Hematology and Oncology, Children's Minnesota, Minneapolis, MN, USA.

An example of a mandibular rhabdomyosarcoma in a 15-year-old male is described featuring EWSR1-TFCP2 fusion with homolateral lymph node metastasis and apparent metastasis to the thoracic vertebra T7. This type of rhabdomyosarcoma has preference for the craniofacial skeleton. Histologically, the tumor was composed of spindle and epithelioid cells characterized by nuclear pleomorphism, cytologic atypia and brisk mitotic activity. Immunohistochemically, it featured diffuse positive nuclear staining MYOD1, only focal staining for myogenin and patchy cytoplasmic staining for desmin. Tumor cells were positive for keratins and nuclear staining for SATB2 was also observed. Interestingly, tumor cells were diffusely positive for calponin. Currently, the patient is under chemotherapy treatment.
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http://dx.doi.org/10.1007/s12105-020-01189-1DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8010041PMC
March 2021

Segmental Ipsilateral Odontognathic Dysplasia (Mandibular Involvement in Segmental Odontomaxillary Dysplasia?) and Identification of PIK3CA Somatic Variant in Lesional Mandibular Gingival Tissue.

Head Neck Pathol 2021 Mar 4;15(1):368-373. Epub 2020 Jun 4.

Division of Oral and Maxillofacial Pathology, School of Dentistry, University of Minnesota, 515 Delaware Street SE #16-116B, Minneapolis, MN, 55455, USA.

Segmental odontomaxillary dysplasia (SOD) is a developmental condition of the middle and posterior maxilla featuring dysplastic bone overgrowth, dental abnormalities and, occasionally, various homolateral cutaneous manifestations. Herein, we describe an individual with maxillary abnormality akin to SOD and associated ipsilateral segmental odontomandibular dysplasia. Also, the result of the evaluation of lesional mandibular gingival tissue for overgrowth-related gene variants is reported. An 8-year-old girl presented clinically with congenital maxillary and mandibular alveolar soft tissue enlargement in the area of the premolars. A panoramic radiograph revealed abnormal trabeculation essentially similar to SOD in the maxilla and mandible with congenitally missing maxillary and mandibular first and second premolars and mandibular canines. Diagnostic mandibular bone biopsy was performed and lesional mandibular gingival hyperplastic tissue was obtained for variant analysis of somatic overgrowth genes PIK3CA, AKT1, AKT3, GNAQ, GNA11, MTOR, PIK3R2. Cone beam computerized tomography (CBCT) disclosed osseous abnormalities on the left side of the maxilla and mandible and very mild osseous expansion in the mandible. Histologically, abnormal bone exhibiting prominent reversal lines was present and associated with fibrocollagenous tissue. Genomic DNA analysis disclosed PIK3CAc.1571G>A; pArg524Lys which was seen at a low mosaic level in the blood, indicating a post-zygotic change. Although this case may be a unique disorder, by sharing features with SOD, one can suggest the possibility of mandibular involvement in SOD. The presence of a PIK3CA variant may support the hypothesis that these segmental disorders could be part of the PIK3CA-related overgrowth spectrum.
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http://dx.doi.org/10.1007/s12105-020-01185-5DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8010023PMC
March 2021

Plasmacytoid cells in salivary pleomorphic adenoma: an alternative interpretation of their immunohistochemical characteristics highlights function and capability for epithelial-mesenchymal transition.

Oral Surg Oral Med Oral Pathol Oral Radiol 2019 Nov 7;128(5):515-529. Epub 2019 Feb 7.

Division of Oral and Maxillofacial Pathology, School of Dentistry, University of Minnesota, Minneapolis, MN, USA; Department of Biochemistry, Molecular Biology and Biophysics, College of Biological Sciences, University of Minnesota, Minneapolis, MN, USA.

Objectives: Plasmacytoid cells (PLCs) in salivary pleomorphic adenoma (SPA) are regarded as modified neoplastic myoepithelia and define plasmacytoid myoepithelioma (pMYO). However, histochemically, immunohistochemically and ultrastructurally, PLCs fail to demonstrate frank myogenous properties. Epithelial-mesenchymal transition (EMT) may explain the phenotypes in SPA. Our aim was to evaluate (1) PLCs with accepted or purported myoepithelial and EMT-related markers; and (2) pMYOs for PLAG1 aberrations by using fluorescence in situ hybridization.

Study Design: Eight SPAs with or without PLC-predominance and 3 pMYOs were immunohistochemically studied.

Results: PLCs in SPA and pMYO exhibited strong, scattered to diffuse positivity for K7, rare K14 positivity and were mostly negative for α-smooth muscle actin, h-caldesmon, and p63/p40. S100 staining was strong and diffuse, whereas calponin was variable. DOG1 was negative. PLCs in pMYO and PLC-rich SPA exhibited selective or diffuse WT1 and D2-40 immunoreactivity. EMT markers SNAIL/SLUG exhibited strong and variable immunoreactivity in PLCs in contrast to weak or absent E-cadherin expression. SOX10 was diffusely and strongly positive. PLAG1 rearrangement was present in 1 pMYO.

Conclusions: PLCs mostly fail to express myoepithelial markers; PLCs are neoplastic cells adapting to microenvironmental changes and capable of EMT; and tumors composed solely of PLCs are apparently SPAs depleted of a ductal component.
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http://dx.doi.org/10.1016/j.oooo.2019.01.081DOI Listing
November 2019

Intracellular calprotectin (S100A8/A9) controls epithelial differentiation and caspase-mediated cleavage of EGFR in head and neck squamous cell carcinoma.

Oral Oncol 2019 08 4;95:1-10. Epub 2019 Jun 4.

Department of Diagnostic and Biological Sciences, School of Dentistry, University of Minnesota, Minneapolis, MN 55455, USA. Electronic address:

Objectives: Calprotectin (S100A8/A9) appears to function as a tumor suppressor in head and neck squamous cell carcinoma (HNSCC) and expression in the carcinoma cells and patient survival rates are directly related. We seek to characterize the suppressive role of calprotectin in HNSCC.

Aims: (1) Investigate changes in S100A8/A9 expression as oral carcinogenesis progresses and (2) determine whether intracellular calprotectin can regulate epidermal growth factor receptor (EGFR), a negative prognostic factor, in HNSCC.

Materials And Methods: Using immunohistochemistry (IHC), S100A8/A9 was analyzed in HNSCC specimens (N = 46), including well-differentiated (WD, N = 19), moderately-differentiated (MD, N = 14), poorly-differentiated (PD, N = 5) and non-keratinizing/basaloid (NK/BAS, N = 8), and premalignant epithelial dysplasias (PED, N = 16). Similarly, EGFR was analyzed in HNSCCs (N = 21). To determine whether calprotectin and EGFR expression are mechanistically linked, TR146 HNSCC cells that are S100A8/A9-expressing or silenced (shRNA) were compared for EGFR levels and caspase-3/7 activity using western blotting and immunofluorescence microscopy.

Results: In normal oral mucosal epithelium, S100A8/A9 stained strongly in the cytoplasm and nucleus of suprabasal cells; basal cells were consistently S100A8/A9 negative. In PED and HNSCC, S100A8/A9 expression was lower than in adjacent normal epithelial tissues (NAT) and declined progressively in WD, MD, PD and NK/BAS HNSCCs. S100A8/A9 and EGFR levels appeared inversely related, which was simulated in vitro when S100A8/A9 was silenced in TR146 cells. Silencing S100A8/A9 significantly reduced caspase-3/7 activity, whereas EGFR levels increased.

Conclusions: In HNSCC, S100A8/A9 is directly associated with cellular differentiation and appears to promote caspase-3/7-mediated cleavage of EGFR, which could explain why patients with S100A8/A9-high tumors survive longer.
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http://dx.doi.org/10.1016/j.oraloncology.2019.05.027DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6662626PMC
August 2019

Pale (Clear) Cell Acanthoma of the Palate.

Head Neck Pathol 2020 Jun 22;14(2):535-541. Epub 2019 Jun 22.

Division of Oral and Maxillofacial Pathology, Department of Diagnostic and Biological Sciences, School of Dentistry, University of Minnesota, 515 Delaware Street SE 16-206B, Minneapolis, MN, 55455, USA.

Clear cell acanthoma (CCA), also known as pale cell acanthoma, represents a rare benign epidermal tumor with strong predilection for the lower extremities of middle-aged individuals and no frank gender preference. The etiology of CCA is poorly understood, although a localized psoriasiform reaction is favored. Herein, we report on the clinicopathologic and immunohistochemical features, and HPV status of an apparent example of oral CCA. A 58-year-old female presented with a well-circumscribed, asymptomatic, exophytic, sessile and erythematous nodule of the right hard palate, measuring 0.7 cm in greatest dimension. Microscopically, the lesion featured parakeratosis and acanthosis with neutrophilic microabscesses and broad elongated rete pegs. In areas, spinous epithelial cells exhibited pale or clear cytoplasm without nuclear pleomorphism, mitoses or cytologic atypia. The supporting connective tissue revealed mild chronic inflammation with few scattered neutrophils and numerous capillary vessels. PAS histochemical stain with and without diastase disclosed the presence of cytoplasmic glycogen in the pale cells. The majority of glycogen-rich epithelial cells stained strongly for EMA and were negative for D2-40. Ki-67 immunostaining was confined only to the basal cell layer of the epithelium. A diagnosis of CCA was rendered. The lesion was negative for human papillomavirus (HPV) infection, as assessed by HPV-DNA PCR using the MY09/11 primers for the L1 conserved region, thus HPV infection does not appear to contribute to the pathogenesis of oral CCA. In conclusion, we report an intraoral example of CCA in order to raise awareness about this entity.
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http://dx.doi.org/10.1007/s12105-019-01050-0DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7235129PMC
June 2020

Solitary oral epidermolytic acanthoma: Case report of a rarely diagnosed entity.

Oral Surg Oral Med Oral Pathol Oral Radiol 2019 Dec 31;128(6):e208-e213. Epub 2019 Mar 31.

Associate Professor, Department of Diagnostic and Biological Sciences, Division of Oral and Maxillofacial Pathology, School of Dentistry, University of Minnesota, Minneapolis, MN, USA.

Epidermolytic acanthoma represents a rare localized form of epidermolytic hyperkeratosis, which resembles warty lesions and shows a strong predilection for the genital skin of males. Here, we present an oral solitary epidermolytic acanthoma affecting a 71-year-old Caucasian man. Clinically, the lesion was white, well-circumscribed, and sessile, measuring 2 mm in diameter and located on the posterior mandibular buccal gingiva. Microscopically, pronounced hyperkeratosis and acanthosis, with formation of keratin crypts was observed. Lesional cells of the spinous and granular epithelial layers exhibited prominent intracellular vacuolar degeneration, as well as eosinophilic paranuclear and perinuclear condensations. Intracytoplasmic eosinophilic globules were also seen. No recurrences have been reported. Investigation for low- and high-risk human papillomavirus (HPV) infection failed to reveal positivity for HPV subtypes 6, 11, 16, and 18. Literature review revealed scarce reports of epidermolytic hyperkeratosis-like changes of the oral mucosa associated with malignant neoplasms and inflammatory processes. Epidermolytic acanthoma should be considered in the differential diagnosis of benign epithelial papillomatous lesions of the oral cavity.
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http://dx.doi.org/10.1016/j.oooo.2019.03.016DOI Listing
December 2019

Benign and malignant odontogenic neoplasms of the jaws show a concordant nondiscriminatory p63/p40 positive immunophenotype.

Oral Surg Oral Med Oral Pathol Oral Radiol 2018 Dec 5;126(6):506-512. Epub 2018 Sep 5.

Associate Professor, Division of Oral and Maxillofacial Pathology, School of Dentistry, University of Minnesota, Minneapolis, MN, USA.

Objectives: Antibody p40, which recognizes exclusively ΔNp63 but not TAp63, has shown diagnostic utility in salivary gland and sinonasal tract malignancies. Although p63 immunophenotypic characterization of odontogenic lesions has been reported, p40 expression has not been previously studied. We aimed to study p40 immunoreactivity in odontogenic tumors (OTs) and odontogenic cysts (OCs) and to investigate possible discriminatory properties of the combined p63/p40 immunoprofile in OTs and OCs.

Study Design: Fourteen ameloblastomas, 7 adenomatoid odontogenic tumors, 6 calcifying epithelial odontogenic tumors, 1 squamous odontogenic tumor, 4 primary intraosseous odontogenic carcinomas, 5 calcifying odontogenic cysts, 4 glandular odontogenic cysts, 3 odontogenic keratocysts, 3 dentigerous cysts, and 1 each radicular and orthokeratinized cysts were stained for p63 (4A4) and p40 (BC28) antibodies.

Results: Ameloblastoma, adenomatoid odontogenic tumor, calcifying epithelial odontogenic tumor, squamous odontogenic tumor, and primary intraosseous odontogenic carcinoma demonstrated concordant p63+/p40+ immunophenotype. P40, similar to p63, highlighted almost all lesional cells of OTs and, overall, the full thickness of the epithelial lining of the cystic areas of OCs and ameloblastoma. The keratin layer of OKC and the adluminal ductal and mucous cells of GOC were p63-/p40-.

Conclusions: Both ΔNp63 and TAp63 isoforms are present in neoplastic and developmental odontogenic lesions; and p63/p40 immunophenotype is nondiscriminatory pertaining to benign and malignant OTs and OCs.
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http://dx.doi.org/10.1016/j.oooo.2018.08.018DOI Listing
December 2018

Ectomesenchymal Chondromyxoid Tumor: A Neoplasm Characterized by Recurrent RREB1-MKL2 Fusions.

Am J Surg Pathol 2018 10;42(10):1297-1305

Department of Pathology, UT Southwestern Medical Center, Dallas, TX.

Ectomesenchymal chondromyxoid tumor is a rare and benign neoplasm with a predilection for the anterior dorsal tongue. Despite morphologic heterogeneity, most cases are characterized by a proliferation of bland spindle cells with a distinctive reticular growth pattern and myxoid stroma. The immunophenotype of these neoplasms is likewise variable; most cases express glial fibrillary acid protein and S100 protein, with inconsistent reports of keratin and myoid marker expression. The molecular pathogenesis is poorly understood; however, a subset of cases has been reported to harbor EWSR1 gene rearrangement. Following identification of an RREB1-MKL2 fusion gene by RNA Sequencing in an index patient, a retrospective review of additional cases of ectomesenchymal chondromyxoid tumors was performed to better characterize the clinical, immunohistochemical, and molecular attributes of this neoplasm. A total of 21 cases were included in this series. A marked predisposition for the dorsal tongue was confirmed. Most cases conformed to prior morphologic descriptions; however, hypercellularity, hyalinized stroma, and necrosis were rare attributes not previously emphasized. The neoplastic cells frequently coexpressed glial fibrillary acid protein, S100 protein, keratin, smooth muscle actin, and/or desmin; a single case was found to contain significant myogenin expression. An RREB1-MKL2 fusion product was identified in 19 tumors (90%), a single tumor (5%) had an EWSR1-CREM fusion product, and the remaining case lacked any known fusion gene by RNA Sequencing. The latter 2 cases subtly differed morphologically from many in the cohort. This series illustrates that recurrent RREB1-MKL2 fusions occur in most, perhaps all, cases of ectomesenchymal chondromyxoid tumor.
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http://dx.doi.org/10.1097/PAS.0000000000001096DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6133728PMC
October 2018

Clinicopathologic and Molecular Characteristics of Familial Cherubism with Associated Odontogenic Tumorous Proliferations.

Head Neck Pathol 2018 Mar 18;12(1):136-144. Epub 2017 Jul 18.

Division of Oral and Maxillofacial Pathology, School of Dentistry, University of Minnesota, 515 Delaware Street SE 16-206B, Minneapolis, MN, 55455, USA.

Cherubism is a rare autosomal dominant condition affecting the jaws and caused by mutations in the gene encoding for the adapter protein SH3BP2 that maps to chromosome 4p16.3. Cherubism is characterized by symmetrically developing bone lesions in the maxilla and mandible. The lesions have been radiographically and histopathologically well-described. Here, we present a family with cherubism with two of its members featuring odontogenic tumorous proliferations in association with persistent central giant cell lesions (CGCL). Specifically, the proband, a 25-year-old male, developed a radiolucent lesion characterized histologically by central odontogenic fibroma-like proliferation in association with a CGCL component, while his mother, at age 57, was diagnosed with primary intraosseous odontogenic carcinoma with areas of benign fibro-osseous lesions. In both patients the lesions occurred in the anterior mandible and presented with clinical enlargement. The son underwent incisional biopsy and did not have additional treatment. His mother underwent extensive mandibulectomy due to widespread tumor. The son has two affected children with classic cherubism while a third child at age 5, had not shown any features of the disease. Mutation analysis of three affected members resulted in the identification of a heterozygous mutation in SH3BP2 (c.1244G>C; p.Arg415Pro). To the best of our knowledge, association of cherubism with odontogenic neoplastic lesions has hitherto not been reported in the literature, thus suggesting a relationship between cherubism with disturbed odontogenesis.
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http://dx.doi.org/10.1007/s12105-017-0837-7DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5873491PMC
March 2018

Immunohistochemical and histochemical characterization of intraosseous arteriovenous malformations of the jaws: analysis of 16 cases with emphasis on GLUT-1 immunophenotype.

Oral Surg Oral Med Oral Pathol Oral Radiol 2017 Aug 25;124(2):165-174. Epub 2017 Apr 25.

Postdoctoral Fellow, Department of Diagnostic and Biological Sciences, School of Dentistry, University of Minnesota, Minneapolis, Minnesota, USA. Electronic address:

Objectives: Intraosseous vascular lesions of the craniofacial region are rare and may cause diagnostic and therapeutic challenges. The purpose of this study was to characterize 16 cases of intraosseous arteriovenous malformations (AVMs) affecting the jaws.

Study Design: Immunohistochemical evaluation was performed using antibodies against α-smooth muscle actin (α-SMA), desmin, CD31, D2-40, and glucose transporter 1 (GLUT-1). Staining with elastic Verhoeff-Van Gieson and Masson trichrome histochemical stains was also performed.

Results: No gender predilection (female:male ratio = 1:1) was observed, with patients' mean age being 50.8 years (SD of ±13.9; range 28-71 years). Predilection for the mandible was observed (12 of 16 [75%]). Immunohistochemically, diffuse endothelial CD31 staining was noted, and α-SMA strongly highlighted smooth muscle cells and pericytes. Desmin-positive vessels were identified in 9 of 16 AVMs (56.3%). D2-40 was uniformly negative in all specimens. AVMs were negative for GLUT-1 (11 of 14 [78.6%]) except for 2 cases (2 of 14 [14.3%]) exhibiting focal limited cytoplasmic GLUT-1 immunoreactivity. One case was equivocal for GLUT-1. Masson trichrome highlighted smooth muscle cells, and elastic fibers were identified in thick-walled arteries.

Conclusions: AVMs of the jaws generally lack expression of GLUT-1, similar to soft tissue vascular malformations. Clinicoradiographic features of intraosseous AVMs in the present study were consistent with the findings of previous studies, although mean age was higher.
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http://dx.doi.org/10.1016/j.oooo.2017.04.008DOI Listing
August 2017

Orthokeratinized Odontogenic Cyst with an Associated Keratocystic Odontogenic Tumor Component and Ghost Cell Keratinization and Calcifications in a Patient with Gardner Syndrome.

Head Neck Pathol 2017 Jun 8;11(2):240-248. Epub 2016 Aug 8.

Division of Oral and Maxillofacial Pathology, School of Dentistry, University of Minnesota, 515 Delaware Street SE 16-206B, Minneapolis, MN, 55455, USA.

Gardner syndrome (GS) is caused by mutations in the APC and besides adenomatous colorectal polyps includes such manifestations as osteomas, epidermoid cysts (ECs) and occasionally multiple pilomatricomas. More than 50 % of ECs in patients with GS exhibit pilomatricoma-like ghost cell keratinization. The latter may be explained by the fact that the development of both GS and pilomatricoma is driven by activation of the Wnt/β-catenin signaling pathway. A 62-year-old, Caucasian male with history of GS presented with a unilocular, mixed radiopaque/radiolucent mandibular lesion causing divergence and external root resorption of involved teeth. Histopathologically, the lesion was composed of two cystic components, an orthokeratinized odontogenic cyst (OOC) and a smaller one with characteristics of keratocystic odontogenic tumor (KCOT) featuring, focally, ghost cells and an epithelial morule-like structure. Dystrophic calcifications essentially similar to those seen in pilomatricomas were observed in the fibrous connective tissue wall. The KCOT and OOC epithelia revealed strong and diffuse cytokeratin (AE1/AE3) and β-catenin immunoreactivity. CD10 positive immunostaining was seen in the keratin and superficial spinous cell layers in both OOC and KCOT. The intraepithelial and mural ghost cells showed a cytokeratin (+), β-catenin and CD10 (-) immunophenotype. The diagnosis of OOC with ghost cell calcifications in association with KCOT was rendered. The patient was lost to follow-up. Although a coincidental co-existence cannot be excluded, ghost cell calcifications mimicking pilomatricoma-like changes in an unusual odontogenic cyst combining OOC and KCOT features as seen in this patient with GS may be explained by the common molecular mechanisms underlying the pathogenesis of cutaneous pilomatricomas and GS.
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http://dx.doi.org/10.1007/s12105-016-0748-zDOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5429271PMC
June 2017

Response to the commentary on "Maxillary pseudotumor as initial manifestation of von Willebrand disease, type 2: report of a rare case and literature review".

Oral Surg Oral Med Oral Pathol Oral Radiol 2016 Aug 24;122(2):260-1. Epub 2016 May 24.

Division of Oral and Maxillofacial Pathology, School of Dentistry, University of Minnesota, Minneapolis, MN, USA.

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http://dx.doi.org/10.1016/j.oooo.2016.04.019DOI Listing
August 2016

A subset of ectomesenchymal chondromyxoid tumours of the tongue show EWSR1 rearrangements and are genetically linked to soft tissue myoepithelial neoplasms: a study of 11 cases.

Histopathology 2016 Oct 6;69(4):607-13. Epub 2016 May 6.

Division of Oral and Maxillofacial Pathology, School of Dentistry, University of Minnesota, Minneapolis, MN, USA.

Aims: Ectomesenchymal chondromyxoid tumour (ECT) is a rare, benign intraoral neoplasm showing a predilection for the anterior dorsum of the tongue. The World Health Organization includes ECT in the pathological spectrum of soft tissue myoepithelioma. EWS RNA-binding protein 1 gene (EWSR1) rearrangement is found in 45% of cutaneous, soft tissue and bone myoepithelial neoplasms, and pleomorphic adenoma gene 1 (PLAG1) aberrations are found in 37% of EWSR1-negative soft tissue myoepitheliomas. The aim of this study was to evaluate the presence of EWSR1 and PLAG1 rearrangements in ECTs.

Methods And Results: Eleven formalin-fixed, paraffin-embedded ECTs were evaluated with fluorescence in-situ hybridization probes for EWSR1 (22q12) and PLAG1 (8q12). Among the 11 ECTs tested, three (27.3%) showed EWSR1 rearrangement in >15% of tumour cells, whereas eight (72.7%) cases did not show EWSR1 rearrangement. Eight of nine (89%) ECTs showed gain of EWSR1, probably representing gain of all or part of chromosome 22, in a varying proportion of neoplastic cells ranging between 1.4% and 27.9%. PLAG1 rearrangement was not detected in the successfully hybridized tissue sections (7/11). No correlation was observed between the molecular and histopathological findings, such as morphology of the neoplastic cells, the presence of atypia, and matrical type.

Conclusions: We identified EWSR1 rearrangement in >25% of ECTs. These results suggest that some ECTs are at least genetically related to myoepithelioma of the soft parts. Finally, PLAG1 aberrations do not appear to be critical in the pathogenesis of ECT of the tongue.
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http://dx.doi.org/10.1111/his.12973DOI Listing
October 2016

Response to the Letter Entitled "Archegonous Cystic Odontoma Is Not Necessarily Primordial" by Ide et al.

Head Neck Pathol 2016 09 8;10(3):422-4. Epub 2016 Feb 8.

Division of Oral and Maxillofacial Pathology, School of Dentistry, University of Minnesota, 515 Delaware Street SE 16-206B, Minneapolis, MN, 55455, USA.

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http://dx.doi.org/10.1007/s12105-015-0679-0DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4972752PMC
September 2016

Clinical utility of myb rearrangement detection and p63/p40 immunophenotyping in the diagnosis of adenoid cystic carcinoma of minor salivary glands: a pilot study.

Oral Surg Oral Med Oral Pathol Oral Radiol 2016 Mar 19;121(3):282-9. Epub 2015 Oct 19.

Division of Oral and Maxillofacial Pathology, School of Dentistry, University of Minnesota, Minneapolis, MN, USA. Electronic address:

Objectives: MYB rearrangement is observed in approximately 28% to 86% of adenoid cystic carcinomas (ACCs). Also, ACC features a p63+/p40+ immunophenotype in greater than 90% of cases, compared with p63+/p40- polymorphous low-grade adenocarcinoma (PLGA). Our aim was to investigate the incidence of (1) MYB rearrangement and (2) p63/p40 immunoreactivity in ACC and PLGA of minor salivary glands (MSGs).

Study Design: Seven cases of ACC as well as five of PLGA were evaluated by using a MYB (6 q23.3) break-apart fluorescence in situ hybridization (FISH) probe. In addition, all cases were immunohistochemically stained with p63 and p40 antibodies.

Results: All five successfully hybridized ACCs featured MYB rearrangement, whereas PLGAs did not show MYB rearrangement. Interestingly, one case of PLGA demonstrated a single intact copy of MYB in greater than 88% of the neoplastic cells. All ACCs exhibited consistent p63+/p40+ staining, whereas PLGAs demonstrated a p63+/p40- immunophenotype.

Conclusions: (1) MYB rearrangement is encountered in ACCs but not PLGAs of MSGs; (2) MYB aberrations, for example, monosomy or deletion, can be seen in PLGAs; (3) combined p63/p40 immunostaining can be used to differentiate ACC from PLGA in incisionally biopsied specimens; and (4) performance of either FISH or p63/p40 immunohistochemistry is expected to be able to confirm the diagnosis of ACC or PLGA in small intraoral biopsies, since both techniques appeared to be diagnostically accurate in this pilot study.
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http://dx.doi.org/10.1016/j.oooo.2015.10.016DOI Listing
March 2016

Maxillary pseudotumor as initial manifestation of von Willebrand disease, type 2: report of a rare case and literature review.

Oral Surg Oral Med Oral Pathol Oral Radiol 2016 Feb 9;121(2):e27-31. Epub 2015 Jun 9.

Division of Oral and Maxillofacial Pathology, School of Dentistry, University of Minnesota, Minneapolis, Minnesota, USA. Electronic address:

Objectives: Von Willebrand disease (VWD) is a bleeding disorder associated with inherited defects of von Willebrand factor (VWF). Type 2 N VWD is characterized by impaired FVIII-binding capacity (VWF:FVIIIB). Pseudotumor (PT) represents a serious complication of hemophilia. Case reports of oral PTs in VWD remain scarce.

Methods: An 11-year-old Caucasian female presented with an expansile gingival mass of the posterior maxilla. Surgical excision of the tumor was performed.

Results: Histopathologically, the tumor was characterized by cystic spaces filled with hemorrhagic material and dense fibrous connective tissue. Postoperatively, the patient suffered prolonged hemorrhage. Results of blood tests showed decreased levels of FVIII function (

Fviii: C) and VWF:FVIIIB. Subsequent gene analysis for type 2 N VWD confirmed heterozygosity for the missense mutation p.Arg816 Trp. A diagnosis of oral PT was rendered.

Conclusions: Here, we report the clinical, radiographic, and microscopic features of a rare example of oral PT leading to the diagnosis of type 2 N VWD.
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http://dx.doi.org/10.1016/j.oooo.2015.05.018DOI Listing
February 2016

Primordial Odontogenic Cyst with Induction Phenomenon (Zonal Fibroblastic Hypercellularity) and Dentinoid Material Versus Archegonous Cystic Odontoma: You Choose!

Head Neck Pathol 2016 Jun 9;10(2):237-44. Epub 2015 Jul 9.

Division of Oral and Maxillofacial Pathology, School of Dentistry, University of Minnesota, 515 Delaware Street SE 16-206B, Minneapolis, MN, 55455, USA.

The most recent A.F.I.P. fascicle defines primordial odontogenic cyst (POC) as a distinct, nonkeratinized, odontogenic cyst of "undetermined origin" forming in the place of a developing normal or supernumerary tooth. However, the majority of examples reported in the literature under this term represent odontogenic keratocysts (keratocystic odontogenic tumors). In addition, there are rare reported cases of cystic odontomas. An 18-year-old Caucasian male presented with a unilocular mandibular radiolucent lesion in the place of a congenitally missing molar. Histologically, it featured nonkeratinizing, thin stratified squamous epithelial lining with areas of spongiosis and foci of vacuolization of individual basal cells without significant nuclear palisading. Focally, budding of the basal cell layer was identified. A zone of increased cellularity featuring induction-type fibroblasts was present subepithelially as well as dentinoid deposits with odontogenic epithelial nests. Immunohistochemically, the epithelial lining was negative for calretinin and the induction-like zone negative for S100 protein, smooth muscle actin, and CD34. The case was externally reviewed by five oral pathologists who provided various diagnostic interpretations including primordial cyst, odontogenic cyst not otherwise specified (NOS), cyst with ameloblastic changes, and unicystic ameloblastoma. At that time, a final diagnosis of odontogenic cyst NOS was rendered with a comment that it may represent a true example of POC or a cystic odontoma. The lesion has not recurred within a 13 year follow-up period after initial excision. An unusual cystic lesion is presented that may represent a true example of POC with dentinoid formation or an archegonous cystic odontoma.
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http://dx.doi.org/10.1007/s12105-015-0640-2DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4838965PMC
June 2016

Recurrent ameloblastoma of the mandible with unusual granular cell component.

Int J Surg Pathol 2015 Jun 11;23(4):298-304. Epub 2015 Feb 11.

University of Minnesota, Minneapolis, MN, USA

Ameloblastomas can present in various clinical and histomorphologic patterns. The granular cell variant accounts for only 3.5% to 5% of ameloblastomas. The aim of this case report is to present an example of ameloblastoma with unusual granular cell component, affecting a 63-year-old woman, in which both the inner and peripheral layers of follicles composed exclusively by eosinophilic granular cells. Assessment of the immunohistochemical and histochemical profile of the lesion was performed and the challenges of such a diagnosis were also addressed.
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http://dx.doi.org/10.1177/1066896915570660DOI Listing
June 2015

An inferior alveolar intraneural cyst: a case example and an anatomical explanation to support the articular theory within cranial nerves.

J Neurosurg 2015 Jun 6;122(6):1433-7. Epub 2015 Feb 6.

Departments of 1Neurosurgery and.

The authors describe the case of an intraneural ganglion cyst involving a cranial nerve (V3), which was found to have a joint connection in support of an articular origin within the cranial nerves. An inferior alveolar intraneural cyst was incidentally discovered on a plain radiograph prior to edentulation. It was resected from within the mandibular canal with no joint connection perceived at surgery. Histologically, the cyst was confirmed to be an intraneural ganglion cyst. Reinterpretation of the preoperative CT scan showed the cyst arising from the temporomandibular joint. This case is consistent with the articular (synovial) theory of intraneural ganglion cysts. An anatomical explanation and potential joint connection are provided for this case as well as several other cases of intraneural cysts in the literature, and thus unifying cranial nerve involvement with accepted concepts of intraneural ganglion cyst formation and propagation.
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http://dx.doi.org/10.3171/2014.12.JNS14798DOI Listing
June 2015

Keratinizing odontogenic cyst with verrucous pattern featuring negative human papillomavirus status by polymerase chain reaction.

Oral Surg Oral Med Oral Pathol Oral Radiol 2015 Apr 18;119(4):e233-40. Epub 2014 Sep 18.

Division of Oral and Maxillofacial Pathology, School of Dentistry, University of Minnesota, Minneapolis, Minnesota, USA. Electronic address:

Verrucous odontogenic cysts (OCs) are extremely rare. Here, we report the clinicopathologic features of this unusual entity and investigate the role of human papillomavirus (HPV) by p16INK4A immunohistochemistry and HPV-DNA polymerase chain reaction (PCR). A 32-year-old male presented with a 8.3 × 4.0 cm, multilocular radiolucency of the left ascending ramus of the mandible. Microscopically, the cystic cavity was lined by hyperplastic stratified squamous epithelium demonstrating marked verrucous morphology with multiple sharp or blunt projections. Mild dysplastic features were also identified. A final diagnosis of keratinizing OC with verrucous hyperplasia and epithelial dysplasia was rendered. Immunohistochemically, the verrucous OC showed foci of moderate-to-intense and diffuse, nuclear, and cytoplasmic p16INK4A positivity as well as weak or absent p53 immunopositivity in the p16INK4A labeled areas. The Ki-67 expression was increased. Interestingly, HPV-DNA PCR failed to reveal transcriptionally active HPV genotypes. Complete surgical excision was performed, with no recurrences seen during a 66-month follow-up.
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http://dx.doi.org/10.1016/j.oooo.2014.09.015DOI Listing
April 2015

Angioimmunoblastic T-cell lymphoma of the oral cavity presenting as gingival mass: report of the histopathologic and molecular characteristics of an unusual case featuring clonal T-cell receptor γ gene rearrangement by polymerase chain reaction.

Oral Surg Oral Med Oral Pathol Oral Radiol 2014 Dec 25;118(6):e198-204. Epub 2014 Jul 25.

Division of Oral and Maxillofacial Pathology, School of Dentistry, University of Minnesota, Minneapolis, MN, USA. Electronic address:

Angioimmunoblastic T-cell lymphoma (AITL) is a rare neoplastic process constituting 15% to 20% of peripheral T-cell lymphomas. We report the clinicopathologic and molecular characteristics of an unusual intraoral manifestation of AITL. A 35-year-old white man with a history of AITL presented with a 2.5-cm, poorly circumscribed, erythematous, exophytic lesion occupying the free and attached buccal gingiva of the right maxillary lateral incisor and canine. Histopathologically, the tumor showed diffuse and intense polymorphic infiltration by small to medium-sized lymphocytes admixed with numerous eosinophils. The neoplastic cells showed strong and diffuse reactivity for CD2, CD3, CD4, CD10, and PD-1 (programmed cell death 1 [PDCD1]). Rare immunopositivity was seen with BCL6 (B-cell CLL/lymphoma 6) and CXCL13 (chemokine [C-X-C motif] ligand 13). Neoplastic cells were negative for CD7 and EBER ISH (Epstein-Barr virus-encoded small RNA in situ hybridization). CD21 did not show any increased follicular dendritic cell component. Polymerase chain reaction-based assay found monoclonal T-cell receptor γ (TRG) gene rearrangements. Diagnosis of recurrent/residual AITL was rendered. Chemotherapy was administered, with the intraoral tumor resolving completely 3 months later.
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http://dx.doi.org/10.1016/j.oooo.2014.07.006DOI Listing
December 2014

Syndromes affecting skin and mucosa.

Atlas Oral Maxillofac Surg Clin North Am 2014 Sep;22(2):135-51

Division of Oral and Maxillofacial Pathology, School of Dentistry, University of Minnesota, 515 Delaware Street SE #16-116B, Minneapolis, MN 55455, USA. Electronic address:

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http://dx.doi.org/10.1016/j.cxom.2014.06.001DOI Listing
September 2014

Fluorescence in-situ hybridization identifies Mastermind-like 2 (MAML2) rearrangement in odontogenic cysts with mucous prosoplasia: a pilot study.

Histopathology 2015 May 23;66(6):791-7. Epub 2015 Jan 23.

Division of Oral and Maxillofacial Pathology, School of Dentistry, University of Minnesota, Minneapolis, MN, USA.

Aims: The pathogenesis of intraosseous mucoepidermoid carcinoma (IMEC) remains unknown. Coexistence with odontogenic cysts (ODC) has been reported in 32-48% of IMEC. Furthermore, prosoplastic mucous cells are often seen in the epithelial lining of ODCs. MECT1-MAML2 fusion transcripts have been identified in >66% of salivary gland MEC cases. The aim of this study was to investigate the presence of MAML2 rearrangement in ODCs featuring mucous prosoplasia.

Methods And Results: Ten cases of ODC with a mucous cell component and three cases of IMEC were evaluated using fluorescence in-situ hybridization. All cases occurred in the mandible. The ODCs exhibited a M:F ratio of 4:1 (mean age 49.2 years), while all IMECs occurred in women (mean age 68.3 years). All three IMECs demonstrated MAML2 rearrangement, in 26-61% of tumour cells. Successful hybridization was observed in nine of 10 cases of ODC. In two of these nine, there was MAML2 rearrangement in 12% and 24% of the lining epithelial cells, while three of the nine showed rearrangement in 7-8% of cells; the remaining four cases were negative.

Conclusions: We identified MAML2 rearrangements in five of nine ODCs lined by mucus-secreting cells. This suggests that at least a subset of ODCs with mucous prosoplasia are characterized by molecular events considered diagnostic for intraosseous and extraosseous MEC.
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http://dx.doi.org/10.1111/his.12526DOI Listing
May 2015

Localized juvenile spongiotic gingival hyperplasia featuring unusual p16INK4A labeling and negative human papillomavirus status by polymerase chain reaction.

J Oral Pathol Med 2015 Jan 28;44(1):37-44. Epub 2014 Jul 28.

Division of Oral and Maxillofacial Pathology, School of Dentistry, University of Minnesota, Minneapolis, MN, USA.

Background: Localized juvenile spongiotic gingival hyperplasia (LJSGH) is a distinct type of gingival hyperplastic lesion with specific clinicopathologic features. Evaluation of the morphological characteristics of LJSGH indicates the potential role of human papillomavirus (HPV) infection as an underlying etiopathogenetic mechanism.

Methods: All cases of LJSGH from 2008 to present were retrieved. Clinical and demographic data were collected. HPV status was investigated by p16INK4A immunohistochemistry and HPV-Polymerase chain reaction (PCR).

Results: Twenty-one cases of LJSGH were identified, 14 (66.7%) affecting males and seven (33.3%) females (M:F = 2:1, age range: 8-36, mean: 13 years). All lesions were well-demarcated, exophytic, erythematous, and hemorrhagic with granular or slightly papillary surface. Preponderance for the maxillary gingiva (19, 90.5%) was observed. Two (9.5%) patients presented with recurrence 20 and 21 months after excision (mean follow-up: 18.7 months). Histopathologically, all LJSGH lesions featured epithelial hyperplasia with intense neutrophilic exocytosis and spongiosis. All cases demonstrated positivity for p16INK4A with the majority of specimens (47.6%) intensely decorated in >50% of the overlying epithelium with focal immunostaining observed in 47.6% and diffuse in 52.4%. Thirteen cases (61.9%) were negative for HPV DNA by PCR, while two (9.5%) were suspicious for the presence of low levels of HPV DNA but definitive genotyping was not possible. One case (4.8%) displayed positivity for HPV-31. The remaining five cases failed the PCR reaction.

Conclusions: Human papillomavirus does not participate in the pathogenesis of LJSGH. P16INK4A expression in the absence of detectable HPV DNA can likely be attributed to the intense inflammation associated with LJSGH.
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http://dx.doi.org/10.1111/jop.12214DOI Listing
January 2015

Nasopharyngeal hyalinizing clear cell carcinoma: report of the histopathologic features of a case showing EWSR1 rearrangements by FISH and literature review.

Int J Surg Pathol 2014 Oct 11;22(7):667-72. Epub 2014 Mar 11.

University of Minnesota, Minneapolis, MN, USA

Background: Hyalinizing clear cell carcinoma (HCCC) is a rare low-grade malignant tumor affecting the minor salivary glands; nasopharyngeal involvement is uncommon.

Methods And Results: A 38-year-old male patient presented with a 3.2 × 4.5 × 4.4 cm expansile mass obliterating the lumen of the nasopharynx and extending into the left nasal cavity. Histopathologically, the tumor was characterized by clear round to polygonal epithelial cells arranged in anastomosing trabeculae and solid nests. The stroma consisted of fibromyxoid connective tissue with areas of intense hyalinization and desmoplasia. Immunohistochemically, strong and diffuse reactivity for AE1/AE3, CK5/6, and p63 was observed. EWSR1 gene rearrangement was confirmed by fluorescence in situ hybridization. The diagnosis of nasopharyngeal HCCC was rendered. Surgical excision was performed along with adjuvant radiotherapy and chemotherapy.

Conclusions: HCCC generally demonstrates good prognosis with low metastatic potential. Identification of EWSR1 gene disruption is usefulin discerning HCCC from other neoplasms with overlapping microscopic features.
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http://dx.doi.org/10.1177/1066896914526778DOI Listing
October 2014

Lymphangioma circumscriptum, angiokeratoma, or superficial vascular ectasia with epithelial hyperplasia?

Oral Surg Oral Med Oral Pathol Oral Radiol 2014 Aug 6;118(2):e53-7. Epub 2013 Dec 6.

Professor, Department of Oral Pathology, Faculty of Dentistry, National and Kapodistrian University of Athens, Athens, Greece.

We report a case of lymphangioma circumscriptum (cavernous lymphangioma with epithelial hyperplasia) in a 12-year-old girl, presenting as a papillary tumor on the right dorsal side of her tongue. Microscopic examination found cavernous vascular channels lined by a single layer of CD31(+), podoplanin-positive, CD34(-) endothelial cells that occupied the papillary lamina propria and were accompanied by epithelial hyperplasia. A review of the literature on oral vascular tumors with epithelial hyperplasia, namely, lymphangioma circumscriptum and angiokeratoma, provided information that draws into question the use of these terms.
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http://dx.doi.org/10.1016/j.oooo.2013.12.003DOI Listing
August 2014

Junctional spitz tumor (nevus) of the upper lip.

Head Neck Pathol 2014 29;8(3):354-8. Epub 2013 Nov 29.

Department of Oral Pathology and Medicine, Dental School, National and Kapodistrian University of Athens, Thivon 2, Goudi, 11527, Athens, Greece,

Spitz tumor (nevus) is a rare, melanocytic lesion that primarily affects children and adolescents. The lesion may share many clinical and histological similarities with malignant melanoma and careful microscopic examination is required to render the proper diagnosis. Complete excision with close follow-up is considered the appropriate treatment. Despite the common presentation of Spitz nevi on the facial skin, lip involvement has only rarely been described in the English language literature. In the present study, a case of Spitz tumor located on the vermilion border of the upper lip in a 16-year old female patient is reported.
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http://dx.doi.org/10.1007/s12105-013-0510-8DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4126917PMC
April 2015

Polymorphous low-grade adenocarcinoma of the upper lip with metachronous myoepithelioma of the buccal mucosa.

Oral Surg Oral Med Oral Pathol Oral Radiol 2014 Jun 21;117(6):e441-8. Epub 2013 Nov 21.

Division of Oral and Maxillofacial Pathology, School of Dentistry, University of Minnesota, Minneapolis, MN, USA. Electronic address:

Examples of multiple minor salivary gland tumors, synchronous or metachronous, are uncommon. We report a patient who initially presented with polymorphous low-grade adenocarcinoma (PLGA) and subsequently with myoepithelioma. A 91-year-old white woman presented in 2009 with a 1-cm, firm, nontender, well-circumscribed nodule of the left side of the upper lip extending to the anterior buccal mucosa. Excisional biopsy revealed PLGA. While the margins were positive, further treatment was not recommended due to the patient's age. In 2011, the patient returned with a 1.5-cm, asymptomatic mass of the left buccal vestibule. Excision of the lesion revealed a circumscribed proliferation of epithelioid and plasmacytoid cells arranged in spherical or whorl-like islands and immersed in a mucinous stroma, consistent with myoepithelioma. The PLGA recurred 3 years after initial diagnosis. Excision was again associated with positive margins, and again no further treatment was recommended. A few months later, at a scheduled follow-up appointment, she presented with a painless nodule of the left upper lip, consistent with recurrent PLGA. One month later, the patient died of unrelated causes. We also present a literature review of multiple minor salivary gland tumors.
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http://dx.doi.org/10.1016/j.oooo.2013.08.030DOI Listing
June 2014