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PubMed1976

Melanotic neuroectodermal tumor of infancy: review of the literature and report of case.

A characteristic case of melanotic neuroectodermal tumor of infancy is reported and the literature regarding this lesion is reviewed. The case reported shows the salient features of previously described melanotic neuroectodermal tumors of infancy; it occurs in infants of less than 1 year of age; it most commonly arises from bone (jaws and skull), has distinctive gross and histologic features, is benign, and is cured by total local excision. Since the teeth in the mandible and maxilla are undergoing development about the time that this tumor occurs, it is not surprising to find tooth buds in close proximity to the expanding neoplastic mass. When odontogenic rests are found in association with the lesion, it must be assumed that it is a collision of the two structures and not that the odontogenic epithelium is the origin for the lesion as previously thought. This is especially true since odontogenic epithelium is not found in the tumor when it occurs outside the jaws. This is the type of lesion that will most probably first be noticed by a dentist and should be managed by the oral surgeon. It is important to note that proper advice regarding future dental problems should be given to the parents of the infant. Such possibilities as crowding or absence of the deciduous or permanent teeth, malocclusion, and possible malformation of the remaining maxillary ridge should be explained. Close dental follow-up is mandatory since recurrence is possible.

Female↗
PubMed1983

Melanotic neuroectodermal tumor of infancy. A review of seven cases.

The melanotic neuroectodermal tumor of infancy (MNTI) is a rare, usually benign, pigmented neuroectodermal tumor which most often involves the maxilla. The authors reviewed seven cases of MNTI, with patient ages of our patients ranged from nine weeks to 18 months; six of the seven were less than six months old at initial diagnosis. Four patients were males, and all were white. One tumor each was located in the femur, the temporal bone, and the epididymis; the remaining lesions occurred in the maxilla. Three of the four maxillary tumors recurred locally; the epididymal and femoral tumors metastasized. Two of these cases had unique clinical or pathologic features. The case of the femoral tumor is remarkable in that it is the first reported one of MNTI presenting in a long bone. This tumor was aggressively malignant; within two months after its discovery, a large mass of similar tumor was formed in the pelvis, and the tumor resulted in the patient's death. To the authors' knowledge, the case of the temporal bone tumor is the first one of MNTI in which neuronal differentiation of the neuroblastic cells is convincingly demonstrated. This finding provides additional evidence in support of the neuroectodermal theory of origin of these neoplasms.

Female↗
PubMed1992

[The melanotic neuroectodermal tumor of childhood. Tumor identification with immunohistochemical methods. A case report].

The authors report a case of a melanotic neuroectodermal tumor of infancy occurring in the maxilla of a 6-month-old female infant. The tumor displayed a biphasic cellular pattern with small neuroblast-like cells and large melanocytic cells showing a highly characteristic immunophenotype. Both tumor cell types immunostained for neuron specific enolase (NSE) and vimentin. The melanocytoid cells reacted additionally with the anti-keratin antibody Kl 1 and with HMB 45, an antibody specific for melanocytic antigens. The melanotic neuroectodermal tumor of infancy usually runs a benign course, however, local recurrence and metastasis may occur. The course of the disease cannot be predicted from morphological findings.

Female↗
PubMedDate not supplied

Diagnostic cytologic features of an epididymal melanotic neuroectodermal tumor of infancy present in scrotal fluid: a case report.

BACKGROUND: The cytologic features of melanotic neuroectodermal tumor of infancy (MNTI) have been described in rare cases only, and these reports have been based solely on direct smears obtained from aspirated tumor material or from touch preparations of resected tumors. CASE: We report, to our knowledge, the first documented case of an epididymal MNTI diagnosed from aspirated hydrocele fluid processed using the ThinPrep system (Cytyc Corp., Boxborough, Massachusetts, U.S.A.). CONCLUSION: Based on a 15 year retrospective review of scrotal fluids submitted for cytologic evaluation to our laboratory, MNTI represented the sole neoplasm that was represented in the fluid and could be specifically diagnosed. Compared to the direct smear evaluation in our case and other published reports, the ThinPrep method enables the recognition of both neuroblast-like cells and larger melanin-containing epithelial cells in scrotal fluid, thereby aiding in the specific diagnosis of MNTI and minimizing the risk of misdiagnosing this tumor as a more aggressive neoplasm.

Adolescent↗
PubMed1982

Melanotic neuroectodermal tumor of infancy: a malignant tumor of the femur.

An 18-month-old white girl had a malignant melanotic neuroectodermal tumor of infancy of the left femur. Light microscopic findings of a biopsy specimen of the femoral lesion demonstrated the characteristic histologic features, that is, nests of small cells with hyperchromatic nuclei and scant cytoplasm lying within cleftlike spaces lined by cuboidal, melanin-producing cells. Electron microscopy confirmed both neuroblastic and melanocytic differentiation. Despite radiation and chemotherapy, an extensive pelvic tumor developed, which prompted laparotomy 20 months after the femoral biopsy, The apparently metastatic tumor was largely necrotic, but viable nests of undifferentiated small cells and rare individual melanin-containing cells were identified. The patient died 4 months later, at 3 years of age. This case is of particular importance because it is the first example of this tumor found in a long bone. No similar lesions were seen in 17,000 primary tumors of bone treated and seen in consultation at this institution. Its aggressively malignant clinical course is also unusual; only 6 of the 159 cases of melanotic neuroectodermal tumor of infancy reported to date have expressed malignant behavior.

Female↗
PubMed1979

Melanotic neuroectodermal tumor of infancy (pigmented melanoameloblastoma).

The paper describes a large pigmented maxillary tumor in a 46-day-old infant. The histological examination showed the tumor to be a melanotic neuroectodermal tumor of infancy (MNTI). No raised urinary levels of vanilmandelic acid could be shown. The tumor was surgically removed and during a 5 1/2-year follow-up period there was no clinical evidence of recurrence. The origin, behavior and treatment of these rare tumors are discussed.

Female↗
PubMed1989

Malignant melanotic neuroectodermal tumor arising from the pineal body.

A case of a melanotic neuroectodermal tumor arising from pineal region of a 4-year-old girl is presented. The tumor had spread diffusely to the meninges, consistent with malignant behavior. Histologically, the tumor consisted primarily of epithelial elements arranged in tubules, cords and nests separated by fibrous vascular tissue in addition to a small neuroblastomatous focus. Melanin pigment was frequently observed in the epithelial tumor cells, and melanin-laden macrophages were also often observed. No teratoid elements were found. Immunohistochemically, tumor cells were positive for neuron-specific enolase but were nonreactive for S-100 protein, epithelial membrane antigen, glial fibrillary acidic protein, vimentin, alpha-fetoprotein and human chorionic gonadotrophin. Ultrastructurally, the epithelial nature of the tumor cells could be easily demonstrated. In addition, melanosomes in various stages in maturation were observed, indicating melanogenesis of the tumor. On the basis of the tumor location and the histological similarities previously observed for the fetal pineal body, it is very likely that this melanotic epithelial tumor could have originated from the fetal pineal gland.

Brain Neoplasms↗
PubMed1995

Malignant melanotic neuroectodermal tumor of infancy.

A 2-month-old boy underwent surgery for removal of a right temporal melanotic neuroectodermal tumor of infancy (MNTI). Histologically the tumor tissue showed signs of malignancy. The child was reexamined several times up to the age of 5 years. Neuroradiological evaluation showed no evidence of tumor recurrence or metastases. No resulting handicap was observed during neurological and psychological follow-up examination at the age of 5 years. Our findings confirm that surgical removal as the therapy of choice provides an excellent prognosis for this kind of tumor in spite of its histologically malignant appearance.

Brain↗
PubMed2002

A clinicopathologic and immunohistochemical analysis of melanotic neuroectodermal tumor of infancy.

OBJECTIVE: The purpose of this study was to review the features of 8 cases of melanotic neuroectodermal tumor of infancy (MNTI) of the jaws with respect to the expression of NB84, CD99, PGP 9.5, specific cytokeratins, and Ki-67, markers not previously reported in this entity. STUDY DESIGN: A clinicopathologic and immunohistochemical analysis of MNTIs in 8 children was undertaken. RESULTS: Patients were aged 2(1/2) months to 14 months. Seven were males. Seven lesions affected the maxilla. Microscopically, collections of larger, melanocyte-like cells were admixed with smaller, neuroblast-like cells. All MNTIs contained melanin; although most showed cellular atypia, mitoses were infrequent (<2 per 10 high-power fields). However, in one lesion in which the melanocyte-like cells appeared less differentiated, 7 mitoses per 10 high-power fields were counted. The larger cells expressed cytokeratins 7 (4/8), 8 (8/8), 18 (6/8), and 19 (3/8); PGP 9.5; neuron-specific enolase (6/8); S100; HMB45; and chromogranin A (2/8). The small cells expressed CD56 (7/8), neuron-specific enolase (7/8), synaptophysin (3/8), PGP 9.5 (3/8), and chromogranin A (2/8). No MNTIs expressed NB84. The most mitotically active tumor was the only one to show membrane expression of CD99 (by both cell populations), have a detectable Ki-67-positive fraction (25% in both the large- and small-cell components), behave aggressively, and require bilateral maxillectomy. All other MNTIs responded to local excision, and none metastasized. CONCLUSIONS: Most MNTIs are benign and respond to conservative excision. Histology is an unreliable means of predicting clinical behavior, but this study has identified some morphologic and phenotypic features that may indicate a more aggressive lesion.

12E7 Antigen↗
PubMedDate not supplied

Melanotic neuroectodermal tumor of infancy.

A 6-month-old female presented with a swelling of the left superior maxillary bone. A radiograph revealed a radiolucent lesion in the left anterior maxilla, and CT, showed a solid mass. At surgery, the lesion was a cystic dental tumor of ectopic location which was treated by excision. The pathological diagnosis was melanotic neurorectodermal tumor. This is an uncommon benign tumor of the neural crest origin that occurs mainly in the maxilla (70% of cases) but can occur in other areas such as the skull and the mandible. CT reveals a hyperdense mass and MR shows a hypointense mass on T1-weighted images and an iso-intense mass on T2-weighted images. The evolution is usually benign after surgical removal.

Diagnosis, Differential↗
PubMed1994

Melanotic neuroectodermal tumor of infancy (MNTI) in the epididymis. A case report with immunohistological studies and special consideration of malignant features.

Melanotic neuroectodermal tumors of infancy (MNTI) are uncommon, usually benign neoplasms, most frequently found in the maxilla. These tumors are extremely rare in the epididymis. Only 18 cases with this site of origin are documented. We report on the third epididymal MNTI with some morphological characteristics of malignancy but favorable clinical outcome. The 2 cm large tumor of a 6-month-old male infant showed large epitheloid cells in the center and small neuroblastoma-like cells at the periphery. Despite invasion of lymphatics there is no evidence of relapse or metastases during 4 years of follow-up. Immunohistochemically, the large tumor cells were distinctly positive for cytokeratin, vimentin, GFAP, the melanoma marker NKI-C3, NSE, and S100. The small tumor cells were only slightly positive for GFAP, NKI-C3, NSE, and S100 but they were negative for cytokeratin and vimentin. Neurofilament and chromogranin could not be proved in the tumor.

Biomarkers, Tumor↗
PubMed1992

Melanotic neuroectodermal tumor of the skull and meninges in infancy.

Three cases of melanotic neuroectodermal tumors of infancy are presented. Two were localized on the midline, involving the skull and extending subdurally. One was located on the inner aspect of the dura and developed intracranially. Two had a benign course following gross total removal. One had a malignant course, recurring locally and spreading within the brain. The difficulties of removing these tumors when they are implanted on the midline are stressed. Histological features of prognostic value are pointed out. Further support for neural crest origin of these tumors is given.

Child, Preschool↗
PubMed1986

Malignant melanotic neuroectodermal tumor of infancy: a case report.

We present a case of melanotic neuroectodermal tumor in the maxilla that followed an aggressively malignant course. In the first biopsy the tumor showed a classical histological picture, while in the last biopsy only malignant undifferentiated cells were evident. The tumor recurred twice after excision, and in spite of radiotherapy and various chemotherapeutic measures the patient died within 16 months of onset. Similar cases reported in the literature are discussed.

Child, Preschool↗
PubMedDate not supplied

Melanotic neuroectodermal tumor of infancy: clinical, radiological, and pathological features.

We present a case of a 4-month-old female infant with a maxillary melanotic neuroectodermal tumor of infancy (MNTI) and review the pooled data from previous publications on this entity. The literature to date comprises 378 reported cases from 1918 to the present, from which data on the presence or absence of metastatic disease was available in 311, and on the presence or absence of local recurrence in 165. These pooled data suggest a local recurrence rate of 36% with metastasis occurring in 7% of cases. At present, the optimal management includes complete surgical excision with clear margins, but there are no reliable histopathological or molecular features to predict the biological behavior in individual cases.

Antigens, Neoplasm↗