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      Cytopathological findings of proliferating pilomatricoma misdiagnosed as a malignant parotid gland tumor

      case-report

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          Abstract

          Background

          Pilomatricoma is a relatively common benign cutaneous adnexal neoplasm with differentiation towards the hair matrix, inner sheath of hair follicle and hair cortex. Proliferating pilomatricoma is a rare variant of pilomatricoma that can rapidly increase and may be misidentified as a malignant tumor. We herein report the cytopathological findings of proliferating pilomatricoma misdiagnosed as a malignant parotid tumor.

          Case presentation

          A 64-year-old man noticed an acne-like nodule in the left parotid region. It was painless, but it increased to a maximum diameter of 4.5 cm over 2 years. Clinically, left parotid gland carcinoma was suspected, and fine-needle aspiration cytology was performed. Clusters of epithelial cells were observed in a necrotic background, and malignant epithelial cells derived from salivary glands were suspected. Histologically, the resected tumor was diagnosed as proliferating pilomatricoma composed of basophilic cells and shadow cells apart from the parotid gland. However, on a re-evaluation of the cytological specimens, the irregular-shaped epithelial cells were considered to be from basophilic cells. Shadow cells with nuclear disappearance were also confirmed. Tumor recurrence and metastasis have not been observed in the four years since surgery.

          Conclusion

          The present case was first interpreted as a malignant parotid gland tumor, but it was actually a benign skin appendage tumor. Pilomatricoma sometimes rapidly increases and may be mistaken for a malignant tumor. Although it is critical to recognize not only basophilic cells but also shadow cells, it cannot be diagnosed by cytological findings. The final diagnosis should be made on excision specimen only.

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          Most cited references7

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          Morphological stages of pilomatricoma.

          In 1983, Ackerman proposed that pilomatricoma represents an infundibular-matrix cyst in its early stages. To study the evolution of this cystic neoplasm, we examined 118 lesions from 116 patients with pilomatricoma histopathologically and categorized the lesions into four distinct and chronological stages: early, fully developed, early regressive, and late regressive. Early lesions (eight cases) were small cystic structures lined by squamoid and basaloid epithelium containing keratin filaments and faulty hair matrix material composed of shadow cells. Fully developed lesions (27 cases) were large neoplasms lined by basaloid epithelium at their periphery, and within, composed of irregularly shaped, densely packed zones of cornified masses containing shadow cells. Early regressive lesions (37 cases) had no apparent epithelial lining but did have basaloid cell foci at the periphery; within, they were composed of pink hair matrix material with shadow cells surrounded by granulation tissue with inflammatory infiltrate and multinucleated histiocytic giant cells. Late regressive lesions (42 cases) had no epithelial component and were composed of irregularly shaped, partially confluent masses of faulty hair material, and calcified (and sometimes metaplastically ossified) shadow cells embedded in a desmoplastic stroma, with little or no inflammatory infiltrate. In four cases, there was a relatively large dermal nodule composed of several interconnected lobules that consisted largely of basaloid cells with only a few areas of shadow cells. We interpreted these lesions as proliferating pilomatricomas. Based upon our histopathologic findings, we propose that pilomatricomas may be categorized into four distinct morphological stages and that these stages reflect the "life" of a pilomatricoma. Thus, the lesion begins as an infundibular matrix cyst and ends up as a calcified and ossified nodule with no visible epithelial component.
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            Pilomatrixoma as a diagnostic pitfall in fine needle aspiration cytology: a case report.

            Pilomatrixoma (pilomatrixoma, calcifying epithelioma of Malherbe) is a relatively uncommon, benign neoplasm arising from the skin adnexa. The tumor can cause diagnostic difficulty not only for the clinician but also for the cytologist. A 62-year-old woman presented with a right submandibular swelling of 4 months' duration. The clinical findings were highly suspicious for malignancy. A fine needle aspiration biopsy was performed. Three preliminary differential diagnoses were offered: mucoepidermoid carcinoma of the submandibular salivary gland, squamous cell carcinomatous deposit in a submandibular lymph node and calcifying odontogenic tumor. Computed tomography demonstrated no bony lesion. No primary site of squamous cell carcinoma could be identified. An excisional biopsy of the swelling was performed, and the histologic diagnosis of pilomatrixoma was made. The cytologic presentation of pilomatrixoma of the right submandibular region can masquerade as that of a malignant tumor, in this case mucoepidermoid carcinoma, squamous cell carcinoma or odontogenic tumor. This case delineates the cytomorphologic features of pilomatrixoma that may mimic carcinoma.
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              Pilomatrixoma: a potential diagnostic pitfall in aspiration cytology.

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                Author and article information

                Contributors
                81-76-218-8264 , k-nozomu@kanazawa-med.ac.jp
                yama-bu@kanazawa-med.ac.jp
                mariko-n@kanazawa-med.ac.jp
                xinguo@kanazawa-med.ac.jp
                a-shioya@kanazawa-med.ac.jp
                snakada@kanazawa-med.ac.jp
                hminato@ipch.jp
                sohsuke@kanazawa-med.ac.jp
                Journal
                Diagn Pathol
                Diagn Pathol
                Diagnostic Pathology
                BioMed Central (London )
                1746-1596
                28 August 2018
                28 August 2018
                2018
                : 13
                : 65
                Affiliations
                [1 ]ISNI 0000 0001 0265 5359, GRID grid.411998.c, Department of Pathology and Laboratory Medicine, , Kanazawa Medical University, ; 1-1 Daigaku, Uchinada, Ishikawa 920-0293 Japan
                [2 ]ISNI 0000 0001 0265 5359, GRID grid.411998.c, Department of Pathology, , Kanazawa Medical University, ; Kanazawa, Ishikawa Japan
                [3 ]ISNI 0000 0000 9573 4170, GRID grid.414830.a, Department of Diagnostic Pathology, , Ishikawa Prefectural Central Hospital, ; Kanazawa, Ishikawa Japan
                Author information
                http://orcid.org/0000-0003-3626-4633
                Article
                738
                10.1186/s13000-018-0738-4
                6114043
                30153836
                68b32738-8f37-4d5b-a6c4-3413249e54d8
                © The Author(s). 2018

                Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License ( http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

                History
                : 18 May 2018
                : 16 August 2018
                Categories
                Case Report
                Custom metadata
                © The Author(s) 2018

                Pathology
                proliferating pilomatricoma,basophilic cells,shadow cells,cytology
                Pathology
                proliferating pilomatricoma, basophilic cells, shadow cells, cytology

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