Clinical and immunohistochemical study of melanotic neuroectodermal tumor of infancy in the maxilla

Santos, Hellen Bandeira de Pontes; Luna, Aníbal Henrique Barbosa; Góes, Pedro Éverton Marques; Sverzut, Alexander Tadeu; Nonaka, Cassiano Francisco Weege; Alves, Pollianna Muniz

doi:10.1590/s1679-45082018rc4025

Cited by 5 publications

(5 citation statements)

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“…More than 500 cases of MNTI have been reported worldwide by Rachidi et al In addition to the 472 cases reported between 1918 and 2013 as a part of the largest retrospective study, [5] we found 49 cases reported between January 2014 and June 2021 by searching PubMed [3,4,9–32,36–38] . In a systematic review of 472 MNTI cases, [5] most cases developed within 1 year of age, with more than 90% of cases occurring in the craniofacial region, such as the maxilla (60.3%), skull (18.1%), and mandible (10.3%).…”

Section: Discussionmentioning

confidence: 75%

Melanotic neuroectodermal tumor of infancy in the mandible

et al. 2021

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Rationale: Melanocytic neuroectodermal tumor of infancy (MNTI) is a rare benign pigmented neoplasm that arises from the neural crest and has an aggressive growth pattern. It is predominantly seen in infants under 1 year of age, and the most common site of involvement is the maxilla. The currently accepted treatment is removal by surgical resection. Herein, we report a case of MNTI that involved the anterior alveolar ridge of the mandible in a 6-month-old infant. Patient concerns: A case of a 6-month-old male child with a huge mass in the anterior alveolar ridge of the mandible. Diagnosis: The tumor was diagnosed using histopathological and immunohistochemical techniques on the biopsy specimen obtained following incisional biopsy. Based on the findings, a final diagnosis of MNTI was established. Interventions: Radical resection of the tumor was performed, after determining the extent of resection by referring to the mandibular 3D model created using the pre-operative CT data. Outcomes: The postoperative course was uneventful, and no recurrence has been observed to date for more than 4 years after surgery. Lessons: This case emphasizes that early diagnosis and radical surgery are critical to the effective treatment, as MNTI exhibits rapid and destructive growth. It also requires careful and close follow-up because of high recurrence rates.

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Section: Discussionmentioning

confidence: 75%

Melanotic neuroectodermal tumor of infancy in the mandible

et al. 2021

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“…However, all those synonyms are obsolete and not recommended. The name of MNTI is widely accepted now and the tumor is proposed to be of neural crest origin because the tumor produces VMA ( 2 ) and has heterogeneous cellular phenotype microscopically which explains by the mesodermal and ectodermal morphological features displayed by neural crest cells at different stages of their ontogeny ( 13 ).…”

Section: Discussionmentioning

confidence: 99%

“…MNTI is categorized as one type of benign maxillofacial bone and cartilage tumors in 4 th edition of WHO Classification of Head and Neck Tumors. However, radiologically, MNTI always presented with well circumscribed hypodense mass, but may show excessive bone destruction and displacement of dental follicles under CT scan ( 13 ). The present case showed a well-defined, unilocular, osteolytic lesion causing expansion and destruction of the maxillary cortical bone, which was in line with the previous publications.…”

Section: Discussionmentioning

confidence: 99%

Melanotic Neuroectodermal Tumor of Infancy: A Clinicopathological and BRAF V600E Mutation Study of 11 Cases

Xia¹,

Zhang²,

Wang³

et al. 2021

Front. Oncol.

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PurposeTo investigate the clinicopathological features and BRAF V600E mutation of melanotic neuroectodermal tumor of infancy (MNTI).Materials and MethodsEleven cases of MNTI diagnosed at the Department of Oral Pathology were collected. Clinicopathological characteristics were obtained from the medical records. Immunostaining was performed by immunohistochemistry (IHC). Amplification-Refractory Mutation System-qPCR (ARMS-qPCR) and Sanger Sequencing were used to detect BRAF V600E mutation.ResultsOf the 11 cases, 3 cases were female and 8 cases were male. The mean age of the first symptoms was 3.2 months (range: 1 to 6 months). Ten cases (90.9%) located in maxilla but only one (9.1%) in mandible. Most of the cases demonstrated well-defined mass with lytic bone destruction and tooth germ affecting radiologically. Histologically, MNTI was consisted of large polygonal melanin-producing epithelioid cells and small round neuroblast-like cells which arranged in irregular alveolar, tubuloglandular and fissured architecture. The epithelioid cells expressed Vim, Pan-CK, NSE and HMB45, while the smalls cells expressed Syn, NSE and scattered Vim. Most cases showed low Ki-67 index (range: <1% to 50%). None of the MNTI cases showed BRAF V600E mutation. Most cases were treated with enucleation (45.4%) or curettage (36.4%). Among the 11 cases, 6 cases had follow-up information, and 2 cases had recurrence lesions after surgery.ConclusionMNTI, an extremely rare tumor, mainly affects male infants with strong preference for maxilla. Distinct histopathological features and immunohistochemical profile are helpful to distinguish from other melanin-containing tumors and small round cell tumors. No BRAF V600E mutation in MNTI is detected in the present study and needs further investigations. The factors that contribute to the local recurrence of MNTI are controversial, but the close follow-up for the patients is recommended.

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“…Radiotherapy is also used, especially for patients who cannot tolerate chemotherapy. Some pPNET patients undergo surgery after chemotherapy or radiotherapy but cannot receive adjuvant chemotherapy and radiotherapy given that combined therapy will increase the risk of complications [ 7 ].…”

Section: Discussionmentioning

confidence: 99%

Resection and reconstruction of a giant primitive neuroectodermal tumour of the abdominal wall with an ultra-long lateral circumflex femoral artery musculocutaneous flap: a case report

et al. 2021

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Background Primitive neuroectodermal tumours are clinically rare. Here, we report a case of a large peripheral primitive neuroectodermal tumour of the abdominal wall. The defect was reconstructed with the longest lateral circumflex femoral artery musculocutaneous flap reported to date. Case presentation A 15-year-old male suffered rupture and bleeding of an abdominal wall mass with a volume of approximately 23*18*10 cm3, involving the whole layer of the abdominal wall. Pathological examination revealed a peripheral primitive neuroectodermal tumour. The tumour was removed via oncologic resection, and the abdominal wall was reconstructed with a bilateral 44*8 cm2 lateral circumflex femoral artery musculocutaneous flap combined with a titanium polypropylene patch. The patient had smooth recovery postoperative, and the functions of the donor and recipient areas of the flap were not significantly affected. Conclusion In this case report, we describe a rare primitive neuroectodermal tumour of the abdominal wall, which invaded almost the entire abdominal wall due to delay of treatment. After thoroughly removing the tumour, we immediately reconstructed the abdominal wall with an ultra-long lateral circumflex femoral artery musculocutaneous flap and achieved better appearance and function after the operation. This case suggests that we should adopt an integrated scheme of surgery combined with radiotherapy and chemotherapy in the treatment of peripheral primitive neuroectodermal tumours. Under the premise of determining the blood supply, the lateral circumflex femoral artery musculocutaneous flap can be cut to a sufficient length.

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Clinical and immunohistochemical study of melanotic neuroectodermal tumor of infancy in the maxilla

Cited by 5 publications

References 10 publications

Melanotic neuroectodermal tumor of infancy in the mandible

Melanotic neuroectodermal tumor of infancy in the mandible

Melanotic Neuroectodermal Tumor of Infancy: A Clinicopathological and BRAF V600E Mutation Study of 11 Cases

Resection and reconstruction of a giant primitive neuroectodermal tumour of the abdominal wall with an ultra-long lateral circumflex femoral artery musculocutaneous flap: a case report

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