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Zhang Z, Hong X, Wang F, Ye X, Yao YD, Yin Y, Yang HY. Solitary intraosseous neurofibroma in the mandible mimicking a cystic lesion: A case report and review of literature. World J Clin Cases 2023; 11:6653-6663. [PMID: 37900249 PMCID: PMC10600983 DOI: 10.12998/wjcc.v11.i27.6653] [Citation(s) in RCA: 2] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/21/2023] [Revised: 08/15/2023] [Accepted: 08/31/2023] [Indexed: 09/20/2023] Open
Abstract
BACKGROUND Neurofibromas are benign tumors of a neurogenic origin. If these tumors occur without any other signs of neurofibromatosis, they are classified as isolated neurofibromas. Neurofibromas in the oral cavity mostly occur within soft tissues, indicating that solitary intraosseous neurofibromas in the mandible are rare. Due to the absence of specific clinical manifestations, early diagnosis and treatment of these tumors are difficult to achieve. CASE SUMMARY A 37-year-old female patient visited our hospital due to numbness and swelling of the gums in the right lower molar area that had persisted for half a month. The patient's overall condition and intraoral examination revealed no significant abnormalities. She was initially diagnosed with a cystic lesion in the right mandible. However, after a more thorough examination, the final pathological diagnosis was confirmed to be neurofibroma. Complete tumor resection and partial removal of the right inferior alveolar nerve were performed. As of writing this report, there have been no signs of tumor recurrence for nine months following the surgery. CONCLUSION This case report discusses the key features that are useful for differentiating solitary intraosseous neurofibromas from other cystic lesions.
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Affiliation(s)
- Zheng Zhang
- School of Stomatology, Zunyi Medical University, Zunyi 563000, Guizhou Province, China
- Department of Stomatology, Peking University Shenzhen Hospital, Shenzhen 518036, Guangdong Province, China
| | - Xia Hong
- Department of Stomatology, Peking University Shenzhen Hospital, Shenzhen 518036, Guangdong Province, China
| | - Feng Wang
- Department of Stomatology, Peking University Shenzhen Hospital, Shenzhen 518036, Guangdong Province, China
| | - Xin Ye
- Department of Stomatology, Peking University Shenzhen Hospital, Shenzhen 518036, Guangdong Province, China
| | - You-Dan Yao
- Department of Stomatology, Peking University Shenzhen Hospital, Shenzhen 518036, Guangdong Province, China
| | - Ying Yin
- Department of Pathology, Peking University Shenzhen Hospital, Shenzhen 518036, Guangdong Province, China
| | - Hong-Yu Yang
- School of Stomatology, Zunyi Medical University, Zunyi 563000, Guizhou Province, China
- Department of Stomatology, Peking University Shenzhen Hospital, Shenzhen 518036, Guangdong Province, China
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Kamalakaran A, Jayaraman B, Raghavendran S, Thirunavukkarasu R, Ayyappan M, Syed JBAB. Intraosseous Neurofibroma of Mandible in a 5-Year-Old: A Rare Case Report and Review of Literature. J Maxillofac Oral Surg 2022; 21:1336-1342. [PMID: 36896066 PMCID: PMC9989095 DOI: 10.1007/s12663-021-01597-y] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/19/2021] [Accepted: 05/26/2021] [Indexed: 10/21/2022] Open
Abstract
Neurofibromas are benign tumours originating from the peripheral nerve sheath mainly the endoneurium. The lesion can occur in a solitary form or as multiple tumours associated with neurofibromatosis (NF-1), which is also known as von Recklinghausen's disease. Intraosseous neurofibromas are exceedingly rare with only less than 50 cases reported in the literature. We report a case of paediatric neurofibroma of the mandible which is even more rare with only 9 cases reported so far. Hence, systematic and thorough investigations are mandatory to correctly diagnose and plan appropriate treatment for intraosseous neurofibroma because of the rarity of the lesion in the paediatric age group. The clinical presentations, diagnostic challenges and treatment plan with a thorough review of literature have been addressed in this case report. The purpose of this paper is to present a case of pediatric intraosseous neurofibroma and to stress the importance of considering such a rare lesion in the differential diagnosis of jaw lesions, especially in children to reduce functional and aesthetic morbidity.
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Affiliation(s)
- Arunkumar Kamalakaran
- Department of Oral and Maxillofacial Surgery, Tamilnadu Government Dental College and Hospital, Chennai-3, India
| | - Balaji Jayaraman
- Department of Oral and Maxillofacial Surgery, Tamilnadu Government Dental College and Hospital, Chennai-3, India
| | - Supraja Raghavendran
- Department of Oral and Maxillofacial Surgery, Tamilnadu Government Dental College and Hospital, Chennai-3, India
| | - Rohini Thirunavukkarasu
- Department of Oral and Maxillofacial Surgery, Tamilnadu Government Dental College and Hospital, Chennai-3, India
| | - Mariammal Ayyappan
- Department of Oral and Maxillofacial Surgery, Tamilnadu Government Dental College and Hospital, Chennai-3, India
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3
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Behrad S, Sohanian S, Ghanbarzadegan A. Solitary intraosseous neurofibroma of the mandible: Report of an extremely rare histopathologic feature. INDIAN J PATHOL MICR 2021; 63:276-278. [PMID: 32317532 DOI: 10.4103/ijpm.ijpm_28_19] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/19/2023] Open
Abstract
Neurofibroma (NF) is a benign tumor derived from the peripheral nerve sheath. Neurofibromas may present either as solitary lesions or as part of the generalized syndrome of neurofibromatosis or von Recklinghausen's disease of the skin. The intraosseous variant of NF is very rare. We report a case of a 32-year-old female who was diagnosed with a solitary intraosseous neurofibroma of the mandible. The present case is rare with respect to its unique histopathologic feature.
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Affiliation(s)
- Samira Behrad
- Department of Oral and Maxillofacial Pathology, Dental School, Semnan University of Medical Sciences, Semnan, Iran
| | - Shabnam Sohanian
- Department of Oral and Maxillofacial Pathology, Dental School, Semnan University of Medical Sciences, Semnan, Iran
| | - Arash Ghanbarzadegan
- Australian Research Centre for Population Oral Health (ARCPOH), Adelaide Dental School, University of Adelaide, Adelaide, South Australia, Australia
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4
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Tamiolakis P, Chrysomali E, Sklavounou-Andrikopoulou A, Nikitakis NG. Oral neural tumors: Clinicopathologic analysis of 157 cases and review of the literature. J Clin Exp Dent 2019; 11:e721-e731. [PMID: 31598201 PMCID: PMC6776406 DOI: 10.4317/jced.55944] [Citation(s) in RCA: 9] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/29/2019] [Accepted: 07/04/2019] [Indexed: 12/12/2022] Open
Abstract
Background Oral neural tumors (ONTs) are rare lesions and represent reactive or neoplastic proliferations of nerve sheath cells. The purpose of the present study is to report the clinical, demographic and histopathologic features of 157 ONTs diagnosed in a single Oral Pathology Department and review the pertinent literature. Material and Methods 157 cases of ONTs diagnosed during a 44-year period were retrospectively collected and the diagnosis was reconfirmed by studying representative haematoxylin and eosin stained tissue sections. The patients’ gender and age, as well as the main clinical features of the lesions, were retrieved from the biopsy submission forms. Results The 157 ONTs represented approximately 0.4% of 35,590 biopsies accessioned during the study period. They affected 71 male and 86 female patients with a mean age of 38.4±18.8 years. They mainly appeared as asymptomatic nodules of normal or white colour on the tongue, lip mucosa and hard palate. The most common ONT was granular cell tumour (38.9%) followed by neurofibroma (19.7%), schwannoma (15.9%), traumatic neuroma (15.9%), palisaded encapsulated neuroma (8.3%) and nerve sheath myxoma (1.3%). Conclusions This study confirmed the rarity of ONTs. Their clinical characteristics mimic other oral lesions; thus, microscopic examination is the only mean to arrive at a definitive diagnosis. Key words:Oral neural tumors; oral neural neoplasms; neurofibroma; oral neurofibroma; schwannoma; oral schwannoma; neurilemmoma; oral neurilemmoma; granular cell tumor; oral granular cell tumor; traumatic neuroma; oral traumatic neuroma; palisaded encapsulated neuroma; oral palisaded encapsulated neuroma.
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Affiliation(s)
- Paris Tamiolakis
- DDS, MSc, Department of Oral Medicine and Pathology, Department of Dentistry, School of Health Sciences, National and Kapodistrian University of Athens, Greece
| | - Evanthia Chrysomali
- DDS, PhD, Associate Professor, Department of Oral Medicine and Pathology, Department of Dentistry, School of Health Sciences, National and Kapodistrian University of Athens, Greece
| | - Alexandra Sklavounou-Andrikopoulou
- DDS, MSc, PhD, Professor, Department of Oral Medicine and Pathology, Department of Dentistry, School of Health Sciences, National and Kapodistrian University of Athens, Greece
| | - Nikolaos G Nikitakis
- MD, DDS, PhD, Professor, Head of Department of Oral Medicine and Pathology, Department of Dentistry, School of Health Sciences, National and Kapodistrian University of Athens, Greece
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5
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Perineurioma of the Tongue: A Case Report and Review of the Literature. J Oral Maxillofac Surg 2019; 77:329.e1-329.e7. [DOI: 10.1016/j.joms.2018.09.032] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/03/2018] [Revised: 09/26/2018] [Accepted: 09/26/2018] [Indexed: 11/18/2022]
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6
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da Silva LP, Gonzaga AKG, Santana T, Sena DAC, de Souza LB. Solitary intraosseous neurofibroma: Report of a rare entity. JOURNAL OF ORAL AND MAXILLOFACIAL SURGERY, MEDICINE, AND PATHOLOGY 2018. [DOI: 10.1016/j.ajoms.2018.06.001] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 10/14/2022]
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7
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Mattsson U, Lindberg P. Oral focal mucinosis of the tongue: A rare clinical entity? ORAL AND MAXILLOFACIAL SURGERY CASES 2017. [DOI: 10.1016/j.omsc.2017.01.006] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/20/2022] Open
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8
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Gomes da Silva W, Martínez MM, Miranda ÁMMA, Silva RB, da Silveira HM, de Almeida OP, Pires FR. Oral perineurioma: clinicopathologic features from two cases and review of literature. Oral Surg Oral Med Oral Pathol Oral Radiol 2017; 123:e91-e98. [DOI: 10.1016/j.oooo.2016.09.223] [Citation(s) in RCA: 10] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/04/2015] [Revised: 09/12/2016] [Accepted: 09/30/2016] [Indexed: 01/29/2023]
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9
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Pummi KP, Aho HJ, Laato MK, Peltonen JTK, Peltonen SA. Tight Junction Proteins and Perineurial Cells in Neurofibromas. J Histochem Cytochem 2016; 54:53-61. [PMID: 16087703 DOI: 10.1369/jhc.5a6671.2005] [Citation(s) in RCA: 20] [Impact Index Per Article: 2.2] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/26/2023] Open
Abstract
Cutaneous neurofibromas consist of axonal processes, Schwann cells, fibroblasts, perineurial cells, mast cells, and abundant extracellular matrix. The distribution and role of perineurial cells in neurofibromas has been uncertain, partly because there has not been a specific immunohistochemical marker for perineurial cells. In this study, tight junctions (TJs) of 16 neurofibromas from 12 patients with neurofibromatosis type 1 (NF1) were analyzed using electron microscopy, immunohistochemistry, and Western transfer analysis. Cell-cell contacts with typical ultrastructural morphology of TJs were seen between adjacent perineurial cells surrounding the small nerves and between contacting perineurial cell processes embedded in tumor stroma. Immunohistochemistry showed expression of claudin-1, claudin-3, and ZO-1 in the intercellular junctions of a subpopulation of tumor cells. Occludin was present mainly in perineurium and claudin-5 localized to the blood vessels. Double immunolabelings were used to identify the cell types expressing claudin-1. The results showed that claudin-1 positive cells were also positive for type IV collagen and epithelial membrane antigen but not for S-100 protein. This labeling pattern is consistent with perineurial cell phenotype. Using claudin-1 as a marker, our results showed that clusters of perineurial cells are distributed around the rudimentary nerves within cutaneous neurofibromas and at the periphery of some neurofibromas.
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Affiliation(s)
- Kati P Pummi
- Department of Medical Biochemistry and Molecular Biology, Turku University Hospital, Turku, Finland
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10
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Jangam SS, Ingole SN, Deshpande MD, Ranadive PA. Solitary intraosseous neurofibroma: Report of a unique case. Contemp Clin Dent 2014; 5:561-3. [PMID: 25395781 PMCID: PMC4229774 DOI: 10.4103/0976-237x.142833] [Citation(s) in RCA: 11] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/04/2022] Open
Abstract
Neural tumors located centrally in jaw bones are relatively rare compared with soft tissue neurofibromas. Less than 50 cases have been reported in the literature with a predilection for mandible. This article aims to elucidate a unique case of intraosseous neurofibroma of mandible in a 62-year-old edentulous female patient associated with facial asymmetry due to the swelling extending from the right body of mandible to left body of mandible. The uniqueness of this case is related to the age and extensiveness of this lesion. A review of clinical, radiographic, histological, and immunohistochemical features, and the surgical management pertaining to this case are discussed along with a review of the literature.
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Affiliation(s)
- Sagar Satish Jangam
- Departments of Oral and Maxillofacial Surgery, Nair Hospital Dental College, Mumbai, Maharashtra, India
| | - Snehal Nilesh Ingole
- Departments of Oral and Maxillofacial Surgery, Nair Hospital Dental College, Mumbai, Maharashtra, India
| | - Mohan Devidas Deshpande
- Departments of Oral and Maxillofacial Surgery, Nair Hospital Dental College, Mumbai, Maharashtra, India
| | - Pallavi Adinath Ranadive
- Departments of Oral and Maxillofacial Surgery, Nair Hospital Dental College, Mumbai, Maharashtra, India
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11
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Solomon LW, Magliocca KR, Going RE, Müller S. Unilocular radiolucency of the mandible. Oral Surg Oral Med Oral Pathol Oral Radiol 2014; 117:397-401. [DOI: 10.1016/j.oooo.2012.06.021] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/30/2012] [Revised: 06/06/2012] [Accepted: 06/29/2012] [Indexed: 10/27/2022]
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12
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Komorski J, Petz Ł, Nienartowicz J, Pałka Ł. Neurofibroma of sinus maxillae. Otolaryngol Pol 2014; 68:94-8. [PMID: 24629742 DOI: 10.1016/j.otpol.2012.07.001] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/19/2012] [Revised: 07/01/2012] [Accepted: 07/23/2012] [Indexed: 10/28/2022]
Abstract
Neurofibroma is a benign tumour composed from Schwann cells. Localization in sinus maxillea is very rare. Authors presenting case which was treatment in Maxillo-Facial Surgery Clinic in Wrocław.
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Affiliation(s)
- Józef Komorski
- Klinika Chirurgii Szczękowo-Twarzowej, Akademicki Szpital Kliniczny, Kierownik Kliniki: Dr hab. n.med. Hanna Gerber, Wrocław, Poland
| | - Łukasz Petz
- Klinika Chirurgii Szczękowo-Twarzowej, Akademicki Szpital Kliniczny, Kierownik Kliniki: Dr hab. n.med. Hanna Gerber, Wrocław, Poland.
| | - Jan Nienartowicz
- Klinika Chirurgii Szczękowo-Twarzowej, Akademicki Szpital Kliniczny, Kierownik Kliniki: Dr hab. n.med. Hanna Gerber, Wrocław, Poland
| | - Łukasz Pałka
- Klinika Chirurgii Szczękowo-Twarzowej, Akademicki Szpital Kliniczny, Kierownik Kliniki: Dr hab. n.med. Hanna Gerber, Wrocław, Poland
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13
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Shetty B, Umesh Y, Kranti K, Seshan H. Periodontal manifestations of von Recklinghausen neuro fibromatosis. J Indian Soc Periodontol 2013; 17:253-6. [PMID: 23869137 PMCID: PMC3713762 DOI: 10.4103/0972-124x.113092] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/05/2011] [Accepted: 02/24/2013] [Indexed: 11/20/2022] Open
Abstract
Neurofibroma is an uncommon benign tumor of the oral cavity derived from the cells that constitute the nerve sheath neurofibromatosis type 1 (NF1), also known as von Recklinghausen's disease, is the most common type of neurofibromatosis and accounts for about 90% of all cases. It is one of the most frequent human genetic diseases, with the prevalence of one case in 3,000 births. Neurofibroma is seen either as a solitary lesion or as part of the generalized syndrome of neurofibromatosis. The solitary form does not differ from the disseminated form or the multiple form of the disease, except that systemic and hereditary factors present in the disseminated form are absent in the solitary type. Oral cavity involvement by a solitary and peripheral plexiform neurofibroma in patients with no other signs of neurofibromatosis is uncommon. The expressivity of NF1 is extremely variable, with manifestations ranging from mild lesions to several complications and functional impairment. Oral manifestations can be found in almost 72% of NF1 patients. This is a case report of a 40-year-old lady with a history of multiple faint rounded densities in the skin, chest pain occasionally since 8 months and breathlessness since 1 year and swelling of the right side of the angle of the mandible with limited mouth opening.
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Affiliation(s)
- Bhavya Shetty
- Department of Periodontics, M.S. Ramaiah Dental College and Hospital, Bangalore, Karnataka, India
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14
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Perineurial Cells in Granular Cell Tumors and Neoplasms With Perineural Invasion. Am J Dermatopathol 2012; 34:800-9. [DOI: 10.1097/dad.0b013e31824ba93b] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/20/2023]
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15
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Kawakami F, Hirose T, Kimoto A, Komori T, Itoh T. Plexiform perineurioma of the lip: A case report and review of literature. Pathol Int 2012; 62:704-8. [DOI: 10.1111/j.1440-1827.2012.02857.x] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/30/2022]
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16
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Abstract
The neurofibroma is a benign tumor of neural origin, not frequently located in the oral cavity, and, especially, extremely rare in the soft palate. When it occurs solitarily, it is a spontaneous tumor without any internal manifestations. We report a case of an isolated neurofibroma of the soft palate not associated with von Recklinghausen disease.
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17
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Abstract
Neurofibromatosis is a genetically-inherited disorder of the nervous system that primarily affects the development and growth of neural (nerve) cell tissues and also causes cafe-au-lait spots on the skin, dysplastic abnormalities of the skin, nervous system, bones, endocrine organs and blood vessels. The two major classifications are NF-1, a generalized form, is the commonest and affects peripheral nerve tissues and NF-2, a rare central form, affects the central nervous system. An unusual finding of oral hamartomas may occur as part of NF-1 and here we presented one such rare case of oral hamartomas in a patient with Von-Recklinghausen's disease.
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Affiliation(s)
- C Anand Kumar
- From the Department of Oral Medicine and Radiology, Kanti Devi Dental College and Hospital, Mathura, Uttar Pradesh, India
| | - RC Jagat Reddy
- From the Department of Oral Medicine and Radiology, Kanti Devi Dental College and Hospital, Mathura, Uttar Pradesh, India
| | - Siddarth Gupta
- From the Department of Oral Medicine and Radiology, Kanti Devi Dental College and Hospital, Mathura, Uttar Pradesh, India
| | - Sanjeev Laller
- From the Department of Oral Medicine and Radiology, Kanti Devi Dental College and Hospital, Mathura, Uttar Pradesh, India
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18
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Extraneural Soft Tissue Perineurioma of the Oral Mucosa. J Oral Maxillofac Surg 2011; 69:1678-82. [DOI: 10.1016/j.joms.2009.07.101] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/19/2009] [Revised: 05/05/2009] [Accepted: 07/27/2009] [Indexed: 11/19/2022]
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19
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Adachi S, Doi R, Mitani K, Iwamoto Y, Furumoto A, Yamashita M, Cho H. Atypical soft tissue perineurioma in the tongue of a young girl. Pathol Int 2010; 60:787-91. [DOI: 10.1111/j.1440-1827.2010.02602.x] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/30/2022]
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20
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Koutlas IG, Scheithauer BW, Folpe AL. Intraoral perineurioma, soft tissue type: report of five cases, including 3 intraosseous examples, and review of the literature. Head Neck Pathol 2010; 4:113-20. [PMID: 20401642 PMCID: PMC2878625 DOI: 10.1007/s12105-010-0177-3] [Citation(s) in RCA: 22] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/27/2009] [Accepted: 03/29/2010] [Indexed: 10/19/2022]
Abstract
Soft tissue type perineuriomas (STP) are uncommon tumors, oral examples being very rare. They have been described in the mandible, gingiva, lips, retrotonsillar mucosa and maxillary vestibule. Herein, we report the clinicopathologic features of five STP, two affecting the buccal mucosa and three the mandible. Three patients were women and two men. All tumors were characterized by a proliferation of cytologically bland, mitotically inactive spindled cells with round, ovoid or spindled nuclei, embedded in a variably collagenous and myxoid matrix. Interestingly, two mandibular tumors featured psammoma bodies and one, in addition, contained irregular calcifications. Tumor cells showed the immunohistochemical profile of perineurial cells including epithelial membrane antigen, Glut-1, claudin-1 and collagen type IV. S100 and neurofilament proteins were not expressed by the tumor cells. A few minute, peripherally situated, entrapped nerves were identified. All tumors were reported gross-totally excised and no recurrences have taken place. The clinical characteristics of STP are summarized and its differential diagnosis relative to other spindle cells tumors and meningioma is discussed.
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Affiliation(s)
- Ioannis G. Koutlas
- Division of Oral and Maxillofacial Pathology, University of Minnesota, School of Dentistry, 515 Delaware Street SE #16-206B, Minneapolis, MN 55455 USA
| | | | - Andrew L. Folpe
- Department of Laboratory Medicine and Pathology, Mayo Clinic, Rochester, MN USA
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21
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Noonan VL, Greene DJ, Brodsky G, Kabani SP. Extraneural sclerosing perineurioma of the buccal mucosa: a case report and clinicopathologic review. Head Neck Pathol 2010; 4:169-73. [PMID: 20364337 PMCID: PMC2878626 DOI: 10.1007/s12105-010-0175-5] [Citation(s) in RCA: 12] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/02/2010] [Accepted: 03/20/2010] [Indexed: 11/25/2022]
Abstract
The perineurioma is an infrequently encountered benign peripheral nerve sheath tumor composed of a clonal proliferation of perineurial cells. Rare cases of perineurioma have been reported in the oral cavity. An extraneural sclerosing perineurioma arising in the buccal mucosa of a 17-year-old male is presented. Histopathologically, the tumor is composed of a well circumscribed nodular proliferation of spindle cells arranged in a storiform growth pattern, in some areas subtly arranged around vascular channels. The tumor cells reveal positive immunostaining for epithelial membrane antigen (EMA), collagen type IV and vimentin, and negative immunostaining for S-100 protein, consistent with a perineurial origin. To the best of our knowledge, this case represents the first report of an extraneural sclerosing perineurioma involving the oral cavity.
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Affiliation(s)
- Vikki L. Noonan
- Department of Pathology, Harvard Vanguard Medical Associates, 133 Brookline Ave., 6th Floor, Boston, MA 02215 USA
| | - David J. Greene
- Oral and Maxillofacial Surgery, Private Practice, Nashua, NH USA
| | - Gilbert Brodsky
- Department of Pathology, Harvard Vanguard Medical Associates, 133 Brookline Ave., 6th Floor, Boston, MA 02215 USA
| | - Sadru P. Kabani
- Department of Pathology, Harvard Vanguard Medical Associates, 133 Brookline Ave., 6th Floor, Boston, MA 02215 USA
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A 38-year review of oral schwannomas and neurofibromas in a Brazilian population: clinical, histopathological and immunohistochemical study. Clin Oral Investig 2010; 15:329-35. [PMID: 20217161 DOI: 10.1007/s00784-010-0389-7] [Citation(s) in RCA: 14] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/20/2009] [Accepted: 02/09/2010] [Indexed: 10/19/2022]
Abstract
The benign peripheral nerve sheath tumours are rare lesions mainly represented by schwannoma and neurofibroma. The present work evaluated the clinical and histopathological features of schwannomas and neurofibromas of the oral cavity diagnosed in a Brazilian population. Among 9.000 cases of oral lesions archived from 1970 to 2008, four schwannomas and 12 neurofibromas were identified, microscopically revised and immunohistochemically certified through a panel including monoclonal antibodies (anti-S100, vimentin, HHF-35 and desmin). From biopsy and histological sections records, clinical and histopathological data were retrieved, reviewed and statistically analysed. Predominantly, schwannomas affected non-white males (3:1), with an age and size averages of 34.7 years and 2.8 cm, respectively. Neurofibromas preferentially occurred on the gingival/alveolar ridge of white females (5:1), with 35.7-year mean age, peak of incidence between 3rd and 5th decade, and size average of 1.7 cm. (12 cases, 75%). The studied tumours exhibited more frequently as a painless, sessile and slow growth very similar to other oral lesions, but their microscopic features differed significantly. Schwannomas and neurofibromas are extremely uncommon in the oral cavity, exhibiting clinical features very similar but specific and peculiar microscopic findings that are useful in the establishment of the diagnosis, which in some particular cases must be confirmed by immunohistochemistry.
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23
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Vencio E, Cheim Jr A, Alencar R, Alencar W, Dias Filho A. Perineurioma of the mandibular dental nerve: a case report and review of the literature. ACTA ACUST UNITED AC 2009. [DOI: 10.1111/j.1752-248x.2009.01037.x] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/28/2022]
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Tanaka A, Alva PG, Miyazaki Y, Yoshida N, Kaneko T, Oku Y, Okamoto E, Sakashita H, Kusama K. Intraneural perineurioma of the tongue: report of a case and review of the literature. ACTA ACUST UNITED AC 2009. [DOI: 10.3353/omp.13.71] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/16/2022]
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Boyanton BL, Jones JK, Shenaq SM, Hicks MJ, Bhattacharjee MB. Intraneural perineurioma: a systematic review with illustrative cases. Arch Pathol Lab Med 2007; 131:1382-92. [PMID: 17824794 DOI: 10.5858/2007-131-1382-ipasrw] [Citation(s) in RCA: 78] [Impact Index Per Article: 4.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 04/23/2007] [Indexed: 01/01/2023]
Abstract
CONTEXT Intraneural perineurioma may be confused with other "onion bulb" Schwann cell entities (localized hypertrophic neuropathy, reactive/demyelinating processes, or inherited polyneuropathies of Charcot-Marie-Tooth/Dejerine Sottas) due to similar clinical, radiologic, and histologic features. Perineurial and Schwann cells can only be differentiated by ultrastructure and immunohistochemsitry. OBJECTIVE To identify and summarize the clinicopathologic features of true cases of intraneural perineurioma from the English language literature. DATA SOURCES A systematic review was performed on definitive intraneural perineuriomas identified through Medline. Baylor College of Medicine-affiliated hospitals' anatomic pathology databases yielded 2 illustrative intraneural perineurioma cases. STUDY SELECTION Intraneural perineurioma inclusion criteria consisted of characteristic histology and confirmation of perineurial cell lineage by either immunohistochemistry (epithelial membrane antigen positive, S100 protein negative) and/or ultrastructural analysis (thin cytoplasmic processes with an incomplete basal lamina, poorly formed tight junctions, and pinocytotic vesicles). DATA EXTRACTION Clinicopathologic data were extracted from all identified articles, with subsequent statistical analysis of the following parameters: age, sex, race, tumor location, tumor size, duration of symptoms prior to diagnosis, treatment modalities and outcomes measures, follow-up assessment for tumor recurrence and metastasis, clinical features (history of trauma, motor/sensory abnormalities, clinical/family history), and diagnostic workup (routine histology, immunohistochemistry, ultrastructural analysis, and molecular/cytogenetic characteristics). CONCLUSIONS Intraneural perineurioma is a neoplastic proliferation of perineurial cells with unique immunohistochemistry and ultrastructural features, and it is distinct from other onion bulb Schwann cell-derived entities. Despite harboring molecular abnormalities of the long arm of chromosome 22, intraneural perineurioma has not been associated with neurofibromatosis. Intraneural perineurioma is a benign peripheral nerve sheath tumor that does not recur or metastasize.
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Affiliation(s)
- Bobby L Boyanton
- Department of Clinical Pathology, William Beaumont Hospital, 3601 W Thirteen Mile Rd, Royal Oak, MI 48073, USA.
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Dundr P, Povýsil C, Tvrdík D, Mazánek J. Intraneural perineurioma of the oral mucosa. Br J Oral Maxillofac Surg 2007; 45:503-4. [PMID: 16757071 DOI: 10.1016/j.bjoms.2006.04.010] [Citation(s) in RCA: 16] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 04/20/2006] [Indexed: 11/29/2022]
Abstract
A 16-year-old boy presented with an intraneural perineurioma arising from a small nerve in the buccal mucosa. Histologically, the tumour comprised a tortuous proliferation of spindle cells arranged like an onion bulb. To our knowledge this is the third example of an intraneural perineurioma that did not present in a major nerve.
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Affiliation(s)
- Pavel Dundr
- Department of Pathology, 1st Medical Faculty, Charles University, Prague, Czech Republic.
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Siponen M, Sándor GK, Ylikontiola L, Salo T, Tuominen H. Multiple orofacial intraneural perineuriomas in a patient with hemifacial hyperplasia. ACTA ACUST UNITED AC 2007; 104:e38-44. [DOI: 10.1016/j.tripleo.2006.12.030] [Citation(s) in RCA: 15] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/08/2006] [Revised: 11/10/2006] [Accepted: 12/21/2006] [Indexed: 10/23/2022]
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Ide F, Mishima K, Yamada H, Saito I, Tanaka A, Kusama K. Neurotized nevi of the oral mucosa: an immunohistochemical and ultrastructural analysis of nevic corpuscles. J Oral Pathol Med 2007; 36:505-10. [PMID: 17686011 DOI: 10.1111/j.1600-0714.2007.00566.x] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/27/2022]
Abstract
BACKGROUND Nevic corpuscle (NC), a stacked lamellar structure reminiscent of Meissner corpuscle, is frequently observed in dermal melanocytic nevi. Although the heading 'neurotized' is classically used for these nevi, the exact neural nature of NC has been a topic of considerable debate. Neurotized nevi have received little attention in the dental literature, and there was no information on NC in oral melanocytic nevi. METHODS Six cases of oral intramucosal nevi with a significant number of NC (two completely and four partially neurotized nevi) were examined immunohistochemically and ultrastructurally. RESULTS NC was composed of closely piled laminar cells devoid of visible melanin. NC and associated spindle nevus cells were immunopositive for S-100 protein but negative for HMB-45, myelin basic protein and epithelial membrane antigen. Within NC, no reactivity for neurofilament protein, protein gene product 9.5 or peripherin was evident. Numerous CD34-positive dendritic cells were located between nevus cells and often encircled NC. Ultrastructurally, NC consisted of concentrically layered elongated cells with a slender lamellated cytoplasm rich in thin filaments and pinocytotic vesicles. Their cytoplasmic processes were focally covered by external basal lamina and continuous to spindle nevus cells. Occasional NC cells contained a few melanosomes. There was no interposed axon in NC. CONCLUSIONS Despite the close resemblance to Meissner corpuscle, NC showed no axonal supply. NC cells lacked terminal Schwannian differentiation and appeared to be modified melanocytes with some perineurial ultrastructural characteristics. The presence of CD34-positive cells, presumably corresponding to endoneurial fibroblasts, further supports an organizational relationship of NC and peripheral nerve sheath elements.
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Affiliation(s)
- F Ide
- Division of Pathology, Department of Diagnostic and Therapeutic Sciences, Meikai University School of Dentistry, Saitama, Japan.
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Ide F, Shimoyama T, Kaneko T, Horie N. Pacinian neuroma in adipose herniation of the buccal mucosa. Int J Oral Maxillofac Surg 2006; 35:1162-3. [PMID: 17008055 DOI: 10.1016/j.ijom.2006.06.025] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/21/2005] [Revised: 04/24/2006] [Accepted: 06/26/2006] [Indexed: 11/23/2022]
Abstract
An interesting case of a trauma-induced tender mass of the buccal mucosa in a 45-year-old man was presented. Following surgery, the patient was relieved from pain. Microscopically, the mature adipose tissue is unique in that it contained a single enlarged Pacinian corpuscle near the deep margin. This is the hitherto undescribed intraoral lesion of Pacinian neuroma in the herniated buccal fat pad.
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Affiliation(s)
- F Ide
- Department of Oral Surgery, Saitama Medical Center, Saitama Medical School, Kawagoe, Japan
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