Kaohsiung Medical University Institutional Repository:Item 310902000/8578

The Journal of Laryngology & Otology September 2001, Vol.115, pp. 763–765

Malignant Žbrous histiocytoma of the tongue

Abstract

A case of malignant Žbrous histiocytoma, which developed in the tongue of a 16-year-old girl is presented. Surgical excision with, or without, radical neck dissection is the treatment of choice of this type of lesion. Key words: Sarcoma; Histocytoma, Fibrous; Tongue

Introduction

Malignant Žbrous histiocytoma (MFH) is a high grade and aggressive sarcoma originally described by O’Brien and Stout in 1964.1_{It is now considered the most common soft}

tissue sarcoma in adults.2 _{Although MFH commonly}

develops in the extremities and retroperitoneum, cases in essentially all regions of the body including the head and neck have been reported. It has been estimated that approximately one to three per cent of these tumours occur in the head and neck region.3 _{Only two cases with}

the tongue as a primary site of MFH have been previously reported.4 ,5 _{The present report describes a further case.}

Case report

A 16-year-old Chinese girl was referred to the Kaohsiung Medical University Hospital due to a rapidly enlarging painless swelling in the left posterior dorsal tongue of Žve days duration (Figure 1). The lesion was neither numb nor tender and was aspirated by a local practitioner but no purulent exudate could be drawn. Her general medical history was unremarkable. Clinical examination revealed a well-deŽned swelling about 23 2.5 cm in diameter, covered with normal appearing mucosa except for a small wound due to aspiration. The swelling was rubbery in consistency and Žxed to the underlying connective

tissue. No limitation of tongue movement was noted. No regional lymphadenopathy was found. A clinical impres-sion of messenchymal tumour was made, however, due to the rapid enlargement of the swelling, malignancy was suspected. The lesion was biopsied and suggested micro-scopically to be giant cell type MFH. She was scheduled for surgical resection of the tumour. A hemiglossectomy with modiŽed radical neck dissection was performed. The margins of the tumour and the neck lymph nodes in the resected specimens were negative for tumour on analysis of frozen sections. The cut surface of the surgical specimen showed a well-delineated tumour surrounded by greyish-white lingual tissue. Histological examination revealed a predominance of multinucleated giant cells, intermixed with Žbroblast-like and histiocyte-like cells (Figure 2), displaying prominent pleomorphism and hyperchromatism (Figure 3). Immunocytochemically, many of the pleo-morphic polygonal cells stained positively with a -1-antic-hymotrypsin but not for keratin, desmin and S-100. Therefore, consistent with the results of the incisional biopsy, a diagnosis of a giant cell type MFH in the tongue

From the Oral Pathology Department, College of Dentistry, Kaohsiung Medical University, Kaohsiung, Taiwan. Accepted for publication: 9 March 2001.

Fig. 1

Swelling over the left dorsal tongue.

Fig. 2

Histological examination revealed a predominance of giant cells, intermixed with fibroblast-like and histiocyte-like cells

(H & E;3 40).

was made. She recovered well post-operatively and remained free of disease at three years follow-up.

Discussion

Although MFH has been accepted as a distinct clinico-pathologic entity, the exact histogenesis of this sarcoma remains uncertain. The majority of investigators have favoured primitive mesenchymal cells such as Žbroblasts and histiocyte-like cells as the origin of these tumours.6

For this case, the histiocyte-like giant cells displayed a -1-antichymotrypsin staining while keratin, desmin and S-100 were all negative. Thus, the diagnosis of MFH was conŽrmed by ruling out other sarcomas, myogenic tumour, sarcomatoid carcinoma or melanoma.

Most reported cases of MFH in the oral region have occurred in bone.7_{Excluding cases of post-radiation MFH}

and cases with the pre-existence of other primary malignancy, only eight cases of MFH arising de novo in the oral cavity as solitary soft tissue lesions have been previously described.1 ,4 ,5 ,8 – 1 1 _{The clinicopathological}

fea-tures of these cases (including the present lesion) are summarized in Table I.

The age of these nine patients at the time of initial treatment ranged from 16 to 73 years with an average of 46 years. The current lesion represents the Žrst such case reported in adolescence. The majority of the patients in this series of MFH were female and the overall male-female ratio was 1:2. The duration of symptoms prior to treatment ranged from Žve days to two years in the seven patients of this series for whom it was reported.5 ,8 – 1 1_The

most common complaint was of a painless enlarging mass, however, in the case reported by Bailey,8 _{a very tender,}

hot and painful lesion was noted. Only two cases in this series (the present case and that reported by Barnes and Kanbour4_{), presented with a rapidly enlarging swelling. At}

the time of initial treatment, the size of the tumours in this series ranged from 2 to 9 cm with an average size of approximately 5 cm.

As can be seen from Table I, the primary treatment of choice in this series of MFH patients was surgical excision with, or without, radical neck dissection. Except for the patient in the case reported by Bailey,8 _{who was observed}

for only a short period, the follow-up period of cases in this series ranged from 18 to 24 months. All patients were alive within 18 months after initial treatment, and only one patient had died of the disease at three years after primary treatment.1 0 _{Furthermore, only two patients had}

recur-rence of the disease, both of which had metastasis to the lung.9 ,1 0 _{It has been suggested that from 25 to 35 per cent}

of patients with MFH of the head and neck will develop metastasis, most often to the lung.8 _{This Žnding suggests}

that the prognosis of patients with solitary oral primary MFH may be good.

Microscopically, MFH can be classiŽed into Žve types: pleomorphic-storiform, giant cell, inammatory, angioma-toid and myxoid.1 0_{Of the nine cases of MFH in this series,}

only storiform-pleomorphic (six patients4 ,5 ,8 ,9 ,1 1_{) and giant}

cell types1 ,1 0 _{were noted. The giant cell type MFH has}

been thought to carry the worse prognosis,1 0 _{however, all}

three cases of giant cell MFH in this series had no evidence of recurrence at a minimum of 18 months follow-up after treatment.

Acknowledgement

The authors wish to acknowledge Dr C. H. Chen, who provided the surgical specimen for this report.

Fig. 3

The giant cells displayed prominent pleomorphism and hyperchromatism (H & E;3 100).

TABLE I

reported cases of primary malignant Ž brous histiocytoma arising solitarily in the oral cavity

Authors Location Age (yr)/sex Histology Symptoms Period beforerecurrence treatmentInitial Follow-up O’Brien and Stout1 _{Buccal mucosa 50/female Giant cell Not}

reported No Excision (24 months)NED Brandenburg and Frank9 _{Lower lip 44/male}

Storiform-pleomorphic reportedNot Yes and lungmetastasis Excision (48 months)LWD DiLascio and Devlin1 1 _{Left cheek 20/female}

Storiform-pleomorphic 8 months No Excision (18 months)NED

Bailey8 _{Upper lip 54/female}

Storiform-pleomorphic 1 month No radiotherapyExcision + NED Manni et al.4 _{Left tongue 61/male}

Storiform-pleomorphic 6 months No Excision +M RND (24 months)NED Bras et al.1 0 _{Lower gingiva 73/female Giant cell 6 months Yes and lung}

metastasis RND (36 months)DOD Barnes and Kanbour4 _{Right hard}

palate 73/female Storiform-pleomorphic 1 month No Excision +RND (48 months)NED Barnes and Kanbour4 _{Right tongue 21/female}

Storiform-pleomorphic 12 months No Excision +M RND (37 months)NED Chen et al. (Present case) Left tongue 16/female Giant cell 5 days No Excision +

M RND (36 months)NED RND = Radical neck dissection; M RND - ModiŽed radical neck dissection; NED = No evidence of disease; LWD = Living with disease; DOD = Died of disease

References

1 O’Brien JE, Stout AP. Malignant Žbrous xanthomas. Cancer1964;17:1445–55

2 Singh B, Shaha A, Har-El G. Malignant Žbrous histio-cytoma of the head and neck. J Craniomaxillofac Surg

1993;21:262–5

3 Weiss SW, Enzinger FM. Malignant Žbrous histiocytoma: an analysis of 200 cases.Cancer1978;41:2250–4

4 Barnes L, Kanbour A. Malignant Žbrous histiocytoma of the head and neck. Arch Otolaryngol Head Neck Surg

1988;114:1149–56

5 Manni JJ, Van Den Broek P, Van Haelst UJ, Slooff JP, Bruaset I. Malignant Žbrous histiocytoma of the tongue. J Maxillofac Surg1986;14:103–7

6 Wood GS, Beckstead JH, Turner RR, Hendrickson MR, Kempson RL, Warnke RA. Malignant Žbrous histio-cytoma tumour cells resemble Žbroblasts. Am J Surg Pathol1986;10:323–35

7 Abdul Karim FW, Ayala AG, Chawla SP, Jing BS, Goepfert H. Malignant Žbrous histiocytoma of the jaws. A clinicopathologic study of 11 cases. Cancer

1985;56:1590–6

8 Bailey BMW. A rare malignant connective tissue tumour arising in the upper lip.Br J Oral Surg1983;21:129–35

9 Brandenburg J, Frank TW. Malignant Žbrous xanthoma of the lip.Otolaryngol Head Neck Surg1980;88:154–6 10 Bras J, Batsakis JG, Lung MA. Malignant Žbrous

histiocytoma of the oral soft tissues.Oral Surg Oral Med Oral Pathol1987;64:57–67

11 DiLascio JP, Devlin GP. Aggressive Žbrous histiocytoma of perioral soft tissues: report of case. J Oral Surg 1981;39:134–6

Address for correspondence: Dr Li-Min Lin,

Oral Pathology and Diagnosis Department, College of Dentistry,

Kaohsiung Medical University, 100, Shih Chuan 1st Road, Kaosiung, Taiwan. Fax: +886 7 3210637

E-mail: [email protected]

Dr Y-K Chen takes responsibility for the integrity of the content of the paper.

Competing interests: None declared