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PubMed2000

Melanotic neuroectodermal tumor of infancy (MNTI) of the hard palate: presentation and management.

OBJECTIVE: To discuss the presentation and management of melanotic neuroectodermal tumor of infancy (MNTI) of the hard palate. METHOD: Case presentation and literature review. CASE: A 6-month-old girl presented with a slow growing, non-tender anterior oral hard palate mass. Radiologic imaging revealed a well-circumscribed cystic lesion containing teeth. After excision, histopathologic and electron microscopic evaluation revealed MNTI. No recurrence was seen at 12-month follow-up. CONCLUSIONS: This case and a review of the literature reveal MNTI to be a rare, benign hard palate tumor, which may present as a smooth, firm, painless, slow-growing anterior palatal lesion. Imaging reveals a well-circumscribed cystic lesion. Complete excision should be curative. Management requires attention to the potential need for palatal reconstruction, orthodontic care and correction of secondary nasal deformities.

Female↗
PubMed1992

Melanotic neuroectodermal tumor of infancy. A case report of paratesticular primary with lymph node involvement.

A 17-month-old boy had a melanotic neuroectodermal tumor of infancy in the left paratesticular region affecting the retroperitoneal lymph nodes. Immunohistochemical and ultrastructural study showed phenotypical diversity of the proliferating cells within a spectrum of neuroectodermal differentiation. Urinary catecholamine levels were initially elevated but returned to normal values after complete eradication of the tumor. The patient received chemotherapy and is now well, without evidence of disease 28 months after surgery.

Biomarkers, Tumor↗
PubMedDate not supplied

Melanotic neuroectodermal tumor of infancy occurring in the left thigh of a 6-month-old female infant.

We report an exceptional case of melanotic neuroectodermal tumor of infancy (MNTI) occurring in the soft tissue of the left thigh of a 6-month-old female infant. The tumor consisted mainly of small round cells (neuroblasts) arranged in cords and nests that were separated by broad fibrovascular areas. In addition, there were a few medium-sized tumor cells containing melanin pigment (melanocytic cells) that in electron microscopy contained melanosomes as well as tonofilaments. Both tumor cell types immunostained for neuron-specific enolase (NSE) and vimentin, and the melanocytic cells reacted additionally with the antikeratin antibody KL1. Within the tumor stroma, neurofilament- and S-100-protein-positive neural cells and vimentin- and desmin-positive myofibroblasts were seen. Although dense-core granules were demonstrated ultrastructurally in some neuroblasts, no immunostaining for chromogranin A, Leu-7, serotonin, or regulatory peptides was found. MNTI located in an extremity can be confused with malignant small round and blue cell tumors of childhood. The distinction between MNTI and these tumors is of clinical significance because MNTI, in most cases, is a benign tumor that, in contrast to the latter, can be cured by complete excision. The presence of a biphasic cell population with neuroblasts and melanocytic cells must be considered the main diagnostic feature of MNTI.

Female↗
PubMed1991

Melanotic neuroectodermal tumor of infancy. A reexamination of a histogenetic problem based on immunohistochemical, flow cytometric, and ultrastructural study of 10 cases.

Ten cases of melanotic neuroectodermal tumor of infancy (MNTI) were studied. There were nine males and one female ranging in age from 2 weeks to 10 months; one patient was 8 years old. Sites of origin were the maxilla (five), epididymis (two), mandible (one), skull (one), and soft tissues of the cheek (one). Six tumors recurred from 1 to 18 months after diagnosis. One patient had widespread dissemination. Electron microscopic study of four cases showed cells with melanosomes at various stages of maturation, and cells with neuroblastic features, including neurosecretory granules and cytoplasmic processes. Nine cases of MNTI were studied immunohistochemically. Small neuroblastic cells and large cells in all cases were reactive for neuron-specific enolase (NSE), synaptophysin, HMB45, and dopamine-beta-hydroxylase, large cells in all cases and few small cells were reactive for cytokeratin (CK) and vimentin (VIM). Epithelial membrane antigen was observed in large cells in three cases, four cases expressed Leu 7 antigen, three were focally positive for glial fibrillary acidic protein, one for desmin, and one for chromogranin. All cases were nonreactive for retinol-binding protein, neurofilaments, alpha-fetoprotein, S-100 protein, and carcinoembryonic antigen. Five normal adult retinas were studied similarly; the pigmented epithelium of the retina was reactive for CK, VIM, HMB45, NSE, and S-100. DNA study, performed in eight tumors, revealed aneuploidy in two (DNA index = 1.7 and 1.8); these cases recurred within 1 month. No differences were observed according to site or behavior. MNTI is a primitive neuroectodermal tumor with polyphenotypic expression of neural and epithelial markers, melanin production, occasional glial, and rhabdomyoblastic differentiation, and no photoreceptor differentiation. It probably represents a dysembryogenetic neoplasm that recapitulates the retina at 5 weeks of gestation.

Child↗
PubMed1986

Tumor of the testicle: a case of melanotic neuroectodermal tumor of infancy.

We describe an 8-month-old boy with a tumor involving the right testicle and epididymis. Anatomical and pathological examination of this tumor revealed embryological tissue and melanin granules derived from the neural crest. This is an uncommon neoplasm, which is especially rare in the testicles. The patient was free of disease after 30 months.

Epididymis↗
PubMed1987

Melanotic neuroectodermal tumor of infancy with high serum levels of alpha-fetoprotein. Ultrastructural study and immunological evidence of glial fibrillary protein and alpha-fetoprotein.

We report an infant melanotic neuroectodermal tumor in the anterior part of the upper gingiva, which was associated with high urinary excretion of vanilmandelic acid. Serum levels of alpha-fetoprotein were abnormally high (200 ng/ml). They returned to normal after removal of the tumor (10 ng/ml). The tumor was characterized histologically and ultrastructurally by the presence of melanocytic and astrocytic cells containing glial fibrillary protein. Intracellular alpha-fetoprotein was demonstrated by the immunoperoxidase method (PAP) in scattered foci of interstitial cells.

Female↗
PubMed1991

Ultrastructural characteristics of a cell line derived from a melanotic neuroectodermal tumor of infancy.

Thin section and freeze-fracture transmission electron microscopy were used to examine and identify the cytoplasmic and membrane structures in a cell line derived from a melanotic neuroectodermal tumor of infancy (MNTI). The cultured cells had a uniform appearance after 70 population doublings characterized by long dendritic processes and evidence of melanin production. The cytoplasm contained numerous melanosomes in various stages of development, vesiculated rough endoplasmic reticulum, microfilaments and uncoated as well as coated vesicles. The membrane specializations included caveoli, coated pits, gap junctions, microfilaments, desmosome-like structures and lamellipodia. The ultrastructural appearance of the cultured MNTI cells was similar to features previously seen in electron micrographs of MNTI tumor specimens. However, correlated freeze-fracture and thin section micrographs permitted further identification of structures previously described. The MNTI cell line represents one of the cell types of the tumor and provides an opportunity for further study of the pathogenesis of this rare tumor.

Cell Line↗
PubMed1999

Cytologic diagnosis of a melanotic neuroectodermal tumor of infancy occurring in the cranial bones.

Melanotic neutroectodermal tumor of infancy (MNTI) is a rare, usually benign tumor commonly occurring in the maxilla. MNTIs at unusual sites like the cranium clinically mimic malignant small round cell tumors. Consequently, a correct preoperative cytologic diagnosis of MNTI at these sites helps in the surgical management of the patient. We report on a cytologically diagnosed case of MNTI in the frontotemporal region of the skull in an infant. Aspirates from the lesion were cellular, with a bimodal population mainly of small neuroblast-like cells admixed with a few large epithelioid cells with melanin granules. In the present case, following the cytologic diagnosis a wide local excision was carried out, and the histologic examination confirmed the cytologic diagnosis. Diagn. Cytopathol. 1999;21:280-283.

Biopsy, Needle↗
PubMed1979

Melanotic neuroectodermal tumor of infancy. An ophthalmic appearance.

Fullness developed in the left side of a 5-month-old male infant's face in the region of the zygoma. An incisional biopsy specimen showed the mass to be a melanotic neuroectodermal tumor, and radical excision was performed. There has been no recurrence of the tumor one year later. Tumors of this type occur in the face, particularly in the maxilla, and have only rarely been reported around the orbit.

Humans↗
PubMed1989

Melanotic neuroectodermal tumor of infancy: a case report.

A male patients, two months of age, was affected by a rapidly growing tumor on the left side of the mandible. There were no other clinical or pathological signs. A thorough examination, as well as radiographic, tomographic and tomodensitometric tests showed the presence of a tumor on the maxilla. It was of low density, encapsulated and shifting the germinal teeth. Surgical removal revealed a melanotic neuroectodermal tumor typical of that in children. The tumor recurred in the same location and was actually larger, less than a month after the operation. A new screening was done for vanilmandelic acid and radiographies of the skull base and skeletal series were negative. The patient was operated on again, with the same histological diagnosis. The diagnosis and treatment are discussed.

Humans↗
PubMed1978

Melanotic neuroectodermal tumor of infancy with highly differentiated neural component. Light and electron microscopic study.

A pigmented tumor was removed from the maxillar alveolar process of a 5-month-old boy. It was examined by light and electron microscopy and a diagnosis of melanotic neuroectodermal tumor was made. In addition to connective tissue, three main cell types were identified: undifferentiated (stem) cells, melanocytes, and nerve cells with processes forming an abundant neuropil. Numerous axo-dendritic and occasional axo-somatic synapses were observed. The neural component demonstrated better differentiation in this example than in any reported so far.

Axons↗
PubMed1992

Melanotic neuroectodermal tumor of infancy: an important mimicker of paratesticular rhabdomyosarcoma.

We report a case of a paratesticular tumor in a 6-month-old infant. This tumor was originally believed to represent a poorly differentiated sarcoma of the cord but upon further pathological consultation it was recognized as a rare melanotic neuroectodermal tumor of infancy involving the epididymis. Since the former is a highly aggressive lesion and the latter an indolent tumor, treatment of these 2 entities differs markedly. Therefore, although the histological distinction between these lesions is often difficult, it is of critical importance.

Diagnosis, Differential↗
PubMedDate not supplied

Melanotic neuroectodermal tumor of infancy initially diagnosed by fine needle aspiration cytology.

A five-month-old male child presented with a tumor of the maxilla, which was clinically diagnosed as an eruption cyst or a rhabdomyosarcoma. Fine needle aspiration smears showed two types of cells: neuroblastlike cells and cells containing melanin pigment. A cytologic diagnosis of melanotic neuroectodermal tumor of infancy was made. This diagnosis was confirmed by histopathologic examination of the subsequently excised mass.

Biopsy, Needle↗
PubMed1981

Melanotic neuroectodermal tumor of infancy and fetal hydantoin syndrome.

Fetal hydantoin syndrome (FHS), a characteristic pattern of altered growth and development, has been well described in recent years in offsprings of epileptic mothers taking phenytoin or other hydantoin anticonvulsants during the gestational period. Recent reports of neuroblastoma in three patients with the FHS further raise the questions of the "oncogenic effect " of hydantoin compounds. A case of melanotic neuroectodermal tumor of infancy (MNTI) has been studied clinically and pathologically including light microscopy, histochemistry, and electron microscopy. This case strengthens the evidence for the teratogenic and oncogenic effects of hydantoin compounds and we believe that it represents the first reported case of FHS associated with MNTI. It would be most important from a clinical standpoint to carefully scrutinize individuals with the FHS for neoplasias. Furthermore, detailed gestational drug history in children with neuroblastoma and other neoplasias should be carefully searched for, with the hope of clarifying the definitive oncogenic effect of hydantoin compounds.

Abnormalities, Drug-Induced↗
PubMed1987

[Intracranial manifestation of so-called "melanotic neuroectodermal tumor of infancy"--a choristoma with neural crest potentials].

A case is reported of so-called "melanotic neuroectodermal tumour of infancy" (= MNTI) in a female newborn. The tumour, whose localization was intracranial, but extracerebral, could be removed subtotally. A further 22 cases of the MNTI with neurocranial manifestations reported in the literature, but differing from our case in their extracranial growth, are listed for comparison. We believe that the neural crest is the histogenic origin of the MNTI and, therefore, our proposed definition is: "Christoma with neural crest potencies."

Brain Neoplasms↗