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Chemotherapy-induced neuronal maturation in sinonasal teratocarcinosarcoma--a unique observation.

Kane SV, Karpate AA, Bal M, Juvekar SL, Pai PS - Head Neck Pathol (2008)

Bottom Line: On imaging, a bone destroying lesion of left paranasal sinuses and nasal cavity was identified.On microscopic examination, it showed similar epithelial and mesenchymal components as the pretreatment biopsies.However, the primitive neuroectodermal component displayed extensive neuronal maturation.

View Article: PubMed Central - PubMed

Affiliation: Tata Memorial Hospital, Parel, Mumbai, Maharashtra, India. drsvkane@gmail.com

ABSTRACT
Sinonasal teratocarcinosarcoma (SNTCS) is a rare and highly malignant tumour with combined features of a teratoma and carcinosarcoma. We report the first case of a SNTCS in 23 year old male treated with neo-adjuvant chemotherapy followed by cranio-facial resection. The resection specimen displayed cellular maturation in the neuroectodermal component. The patient presented with a short history of nasal obstruction, epistaxis and headache. On imaging, a bone destroying lesion of left paranasal sinuses and nasal cavity was identified. The diagnosis of SNTCS could be offered only on the third biopsy which showed heterogeneous admixture of primitive neuroectodermal, epithelial and mesenchymal elements. An adequate sampling with high index of suspicion is needed to catch hold this rare tumor. Tumor was excised after 4 cycles of neo-adjuvant chemotherapy. On microscopic examination, it showed similar epithelial and mesenchymal components as the pretreatment biopsies. However, the primitive neuroectodermal component displayed extensive neuronal maturation. The undifferentiated neuroectodermal cells were completely absent in the post chemotherapy specimen. This case throws light on the morphologic evidence of chemotherapy induced maturation in the neuroectodermal component within SNTCS, an event hitherto not reported in the literature in case of SNTCS.

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a & b Various components of the tumour in the form of heterogeneous admixture of glandular structures, nodules of neural tissue with neurofibrillary matrix and fibroblastic stroma, as seen in the resection specimen. (A-100×, B-200× Hematoxylin & Eosin)
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Fig3: a & b Various components of the tumour in the form of heterogeneous admixture of glandular structures, nodules of neural tissue with neurofibrillary matrix and fibroblastic stroma, as seen in the resection specimen. (A-100×, B-200× Hematoxylin & Eosin)

Mentions: Patient underwent a craniofacial resection for a T3N0 sinonasal tumour following the neo-adjuvant chemotherapy. The specimen was received in multiple, partly hemorrhagic soft-to-rubbery tan-coloured tissue bits, having variegated appearance, the largest one measuring 2.5 × 2.0 × 1.0 cm. The entire tissue was submitted for processing. On microscopy, the tumour was heterogeneous and composed of intimate admixture of neuroectodermal, epithelial and mesenchymal components (Fig. 3). The neuroectodermal component was composed of immature ganglions cells embedded in abundant neurofibrillary matrix (Fig. 4). Although all the 16 hematoxylin and eosin-stained sections exhibited varying proportions of neurofibrillary matrix, it was predominant in the intracranial portion of the tumour. None of the sections showed primitive neuroectodermal cells as seen in the pre-chemotherapy biopsy. Embedded within the neurofibrillary matrix, numerous invasive epithelial islands composed of malignant squamous elements and characteristic hybrid squamo-glandular units were noted. A moderate atypia and mitotic activity were demonstrated in the epithelial elements. Mesenchymal component was composed of myofibroblastic proliferation with collagenized stroma. No obvious sarcomatous component was identified. Bone or cartilage was not seen. Surgical margins were positive for tumour. Scanty uninvolved brain paranchyma was also noted. Mature neuronal component was immunoreactive to neurofilament protein (NFP) [1:100; DAKO] (Fig. 5), NSE [1:100; Dako], synaptophysin [1:100; Dako], chromogranin [1:100; DAKO] and GFAP [1:1200; DAKO]. S-100 protein was focally positive. It was immunonegative for MIC2 [1:100; DAKO]. The glandular epithelial component was immunoreactive to epithelial membrane antigen [1:200; DAKO] and CK7 [1:100; DAKO] while the squamous component was positive for high molecular weight cytokeratin [1:100; DAKO] and p63 [1:200; DAKO] (Fig. 6). Positive labelling with vimentin and smooth muscle actin [1:400; DAKO] was observed in the mesenchymal component. There was no evidence of a germinoma, embryonal carcinoma, yolk-sac tumour or choriocarcinoma in any of the sections.Fig. 3


Chemotherapy-induced neuronal maturation in sinonasal teratocarcinosarcoma--a unique observation.

Kane SV, Karpate AA, Bal M, Juvekar SL, Pai PS - Head Neck Pathol (2008)

a & b Various components of the tumour in the form of heterogeneous admixture of glandular structures, nodules of neural tissue with neurofibrillary matrix and fibroblastic stroma, as seen in the resection specimen. (A-100×, B-200× Hematoxylin & Eosin)
© Copyright Policy
Related In: Results  -  Collection

Show All Figures
getmorefigures.php?uid=PMC2807528&req=5

Fig3: a & b Various components of the tumour in the form of heterogeneous admixture of glandular structures, nodules of neural tissue with neurofibrillary matrix and fibroblastic stroma, as seen in the resection specimen. (A-100×, B-200× Hematoxylin & Eosin)
Mentions: Patient underwent a craniofacial resection for a T3N0 sinonasal tumour following the neo-adjuvant chemotherapy. The specimen was received in multiple, partly hemorrhagic soft-to-rubbery tan-coloured tissue bits, having variegated appearance, the largest one measuring 2.5 × 2.0 × 1.0 cm. The entire tissue was submitted for processing. On microscopy, the tumour was heterogeneous and composed of intimate admixture of neuroectodermal, epithelial and mesenchymal components (Fig. 3). The neuroectodermal component was composed of immature ganglions cells embedded in abundant neurofibrillary matrix (Fig. 4). Although all the 16 hematoxylin and eosin-stained sections exhibited varying proportions of neurofibrillary matrix, it was predominant in the intracranial portion of the tumour. None of the sections showed primitive neuroectodermal cells as seen in the pre-chemotherapy biopsy. Embedded within the neurofibrillary matrix, numerous invasive epithelial islands composed of malignant squamous elements and characteristic hybrid squamo-glandular units were noted. A moderate atypia and mitotic activity were demonstrated in the epithelial elements. Mesenchymal component was composed of myofibroblastic proliferation with collagenized stroma. No obvious sarcomatous component was identified. Bone or cartilage was not seen. Surgical margins were positive for tumour. Scanty uninvolved brain paranchyma was also noted. Mature neuronal component was immunoreactive to neurofilament protein (NFP) [1:100; DAKO] (Fig. 5), NSE [1:100; Dako], synaptophysin [1:100; Dako], chromogranin [1:100; DAKO] and GFAP [1:1200; DAKO]. S-100 protein was focally positive. It was immunonegative for MIC2 [1:100; DAKO]. The glandular epithelial component was immunoreactive to epithelial membrane antigen [1:200; DAKO] and CK7 [1:100; DAKO] while the squamous component was positive for high molecular weight cytokeratin [1:100; DAKO] and p63 [1:200; DAKO] (Fig. 6). Positive labelling with vimentin and smooth muscle actin [1:400; DAKO] was observed in the mesenchymal component. There was no evidence of a germinoma, embryonal carcinoma, yolk-sac tumour or choriocarcinoma in any of the sections.Fig. 3

Bottom Line: On imaging, a bone destroying lesion of left paranasal sinuses and nasal cavity was identified.On microscopic examination, it showed similar epithelial and mesenchymal components as the pretreatment biopsies.However, the primitive neuroectodermal component displayed extensive neuronal maturation.

View Article: PubMed Central - PubMed

Affiliation: Tata Memorial Hospital, Parel, Mumbai, Maharashtra, India. drsvkane@gmail.com

ABSTRACT
Sinonasal teratocarcinosarcoma (SNTCS) is a rare and highly malignant tumour with combined features of a teratoma and carcinosarcoma. We report the first case of a SNTCS in 23 year old male treated with neo-adjuvant chemotherapy followed by cranio-facial resection. The resection specimen displayed cellular maturation in the neuroectodermal component. The patient presented with a short history of nasal obstruction, epistaxis and headache. On imaging, a bone destroying lesion of left paranasal sinuses and nasal cavity was identified. The diagnosis of SNTCS could be offered only on the third biopsy which showed heterogeneous admixture of primitive neuroectodermal, epithelial and mesenchymal elements. An adequate sampling with high index of suspicion is needed to catch hold this rare tumor. Tumor was excised after 4 cycles of neo-adjuvant chemotherapy. On microscopic examination, it showed similar epithelial and mesenchymal components as the pretreatment biopsies. However, the primitive neuroectodermal component displayed extensive neuronal maturation. The undifferentiated neuroectodermal cells were completely absent in the post chemotherapy specimen. This case throws light on the morphologic evidence of chemotherapy induced maturation in the neuroectodermal component within SNTCS, an event hitherto not reported in the literature in case of SNTCS.

Show MeSH
Related in: MedlinePlus