Periosteal fasciitis of the mandible

PERIOSTEAL FASCIlTlS OF THE MANDIBLE

Alan J. Mighell, BChD, Alec S. High, FDS, RCS, MRCPath, and Leo F. A. Stassen, FDS, RCS, FRCS(Ed)

Abstract Background. Periosteal fasciitis is rare in the head and neck. We believe only two cases of fasciitis- induced erosion through the mandibular cortical plate have previously been reported.

Results. Management of a case of periosteal fasciitis with bone erosion at the mandibular angle is reported in a 38- year-old man.

Conclusions. Misdiagnosis of fasciitis in the past has led to inappropriate management and unnecessary morbidity. The principles of diagnosis and management of this unusual and confusing disorder are highlighted.

0 1994 John Wiley & Sons, Inc. HEAD & NECK 1994;16:282-286

T h e differentiation between fasciitis and other soft tissue swellings is essential if the appropriate management is to be implemented. However, ex- cluding infections and cysts, soft tissue swellings of the head and neck are uncommon, especially where associated with bone. When considering a diagnosis of fasciitis the differential diagnosis should include lesions of divergent biologic be-

From the Department of Oral and Facial Surgery (Drs. Mighell and Stas- sen), Sunderland District General Hospital, Sunderland, United Kingdom, and Department of Oral Pathology (Dr. High), Leeds Dental Institute, Leeds, United Kingdom.

Acknowledgments: The authors thank the Medical Illustration Units at Sunderland District General Hospital and Leeds Dental Institute for pro- viding the figures

Address reprint requests to Dr. Mighell at the Department of Oral Pathol- ogy, Leeds Dental Institute, Clarendon Way, Leeds. LS2 9LU UK.

Accepted for publication October 8. 1993

0 1994 John Wiley & Sons, Inc CCC 01 48-6403/94/030282-05

havior, such as fibroma, neurofibroma, neurolem- moma, fibrous histiocytoma, fibromatosis, and many different types of sarcoma.' Despite the clinical and histologic features of fasciitis being well-described, confusion has occurred between fasciitis, a benign reactive condition, and sarcoma, with unnecessary radical surgery, radio- therapy, and chemotherapy.2 This is partially be- cause of the scarcity of fasciitis and sarcoma. Majumdar3 identified 254 cases of head and neck fasciitis in the literature since fasciitis was first described in 1955* and sarcomas represent less than 1% of head and neck neopla~ms.~

CASEREPORT

A 38-year-old white man who was a welder was referred by his general medical practitioner for an urgent opinion regarding an aching pain arising from the right mandibular angle. The swelling had increased in size during the preceding 4 to 5 months, but had only ached intermittently for the previous 2 to 3 weeks. The patient was a non-insulin-dependent diabetic and well con- trolled by diet and oral hypoglycemics.

Extraoral examination identified a nontender, smooth, bony-hard mass approximately 2 cm in diameter attached to the inferior aspect of the right mandibular angle. Overlying skin had a normal appearance, was not attached to the lesion, and there was no cutaneous paraesthesia or facial nerve deficit. No regional lymphadenopathy or evidence of distant disease was detected. The patient was edentulous and intraoral exam-

282 Periosteal Fasciitis of the Mandible HEAD & NECK May/June 1994

ination was unremarkable with no swelling or paraesthesia identified.

Panoramic radiography showed a well- circumscribed and corticated lesion eroding the inferior border of the right mandible. Computed tomography (CT) confirmed these findings and were suggestive of a benign soft tissue mass approximately 2 cm in diameter and eroding mandibular bone (Figure 1). A presumptive diagnosis of a benign connective tissue lesion was made. Initially, an excisional biopsy was planned with an early return to theater if histopathology indi- cated a more aggressive process. Under general anesthesia the right angle of the mandible was approached via a standard submandibular inci- sion. The lesion and a margin of normal bone were removed using burrs. The lesion appeared firm, well-circumscribed from the overlying soft tissue, and possibly encapsulated. Postoperative recovery was unremarkable and at review 1 year later there was no clinical or radiographic evidence of persistent disease.

Histologically, the bulk of the lesion consisted of loosely arranged fascicles of spindle-shaped cells with a vague storiform pattern in places. Scattered osteoclast-like cells and a few lympho- cytes were evident, and, although mitoses were easily found, these were normal. Occasional reactive bone rimmed by plump osteoblasts was

FIGURE 1. Axial CT scan showing replacement of bone at the left mandibular angle by a well-defined lesion (arrow).

present with other areas of more metaplastic os- teoid (Figure 2). Accordingly, it was felt that this was more in keeping with a reactive rather than sarcomatous process, and a diagnosis of periosteal fasciitis was made.

DISCUSSION

Subsequent to the initial report of Konwaler and co-workers4 on pseudosarcomatous fibromatosis (fasciitis), several descriptive terms have been coupled with fasciitis, with nodular fasciitis the most frequently used term. Periosteal fasciitis re- lating to reactive periosteal overgrowth with reactive bone formation or bone destruction was originally reported as parosteal fasciitis and only accounts for 4% of all fasciitis cases.6-8 Similar to periosteal fasciitis but confined to skull bones, cranial fasciitis originates from the cranial peri- osteum and its associated deep f a ~ c i a . ~ Prolifera- tive fasciitis derives from the fascia and fibrous septae of adipose tissue but may occur within fat without fascia1 attachment." Intravascular fasciitis involves predominantly small to medium ar- teries intraluminally, intramurally, and extra- murally. " Proliferative myositis arising from the fascia and septae of skeletal muscle is recognized as a variant of nodular f a s ~ i i t i s . ' ~ ' ~ ~

Fasciitis has been reported in many races in- cluding whites, blacks, Orientals, and American Indians, with an equal sex distribution identified in the larger series.14-17 With the exception of cranial fasciitis, which is a disease of childhood, fasciitis can present a t any age with a peak inci- dence in the fourth

Fasciitis, the most common soft tissue pseudo- sarcoma, mostly presents on the upper extremity followed by the lower extremity and t r ~ n k . ' ~ , ' ~ , ~ ~ Typically, lesions are superficial in subcutaneous tissues, although deep lesions do occur. Head and neck fasciitis accounts for between 7% and 20% of all cases of fasciitis reported, with facial and neck skin being the predominant site^.^-'^ Less common sites include the forehead, scalp, orbit, eye- lid, conjunctiva, parotid sheath, and parotid gland, whereas lesions rarely occur in the trachea, hypopharynx, esophagus, or intra~rally.',~*~,~~,~~-~~ Fasciitis associated with the mandible has been reported at the symphysis, parasymphysis, inferior border, angle, and a l v e o l ~ s . ~ ~ ~ ~ ~ ~ ~ ~ ~ ~ ~ ~ ~ ~ - ~ ~ Clinically, the most striking features are rapid growth of a single, painless or mildly tender, firm nodule without neurologic i n ~ o l v e m e n t . ~ , ~ ~ Mul- tiple lesions or associated lymphadenopathy is unusual .14J9,21 Growth can be so rapid that a sig-

Periosteal Fasciitis of the Mandible HEAD & NECK May/June 1994 283

FIGURE 2. Photomicrograph from the center of the lesion demonstrating spindle-shaped cells and bone (arrow) (hematoxylin & eosin, original magnification ~ 1 6 0 ) .

nificant mass may be apparent within days, although growth can occur for several months as in the case presented. l4 Neurologic involvement only occurs if a rapidly growing lesion presses on a peripheral nerve.’ Most lesions have a diameter of approximately 2 cm, as does this case, although lesions 5 cm x 7 cm have been reported at the anterior mandible and neck.3’7’15,18,45*47

With the exception of cranial fasciitis, reports of fasciitis-induced bony erosion are rare in the head and neck. Sat0 et al.27 describe erosion of maxillary bone, whereas Rakower31 reported ca- nine root resorption in addition to maxillary bone destruction. Rahn and Schneider41 presented a case of mandibular mental region alveolar bone destruction in the German literature. A less ex- tensive area of alveolar bone destruction in the mandibular body was reported by Mostofi et al.39 The case presented adds to these two mandibular cases with erosion through the cortical plate but differs in that the bony destruction occurred at the inferior border of the mandibular angle. Fas- ciitis-induced superficial saucerization with an intact cortical plate has been noted in the mandi- b1e.44,45

Plain radiographs of fasciitis-induced bony destruction tend to have a well-defined margin suggestive of a benign p r o c e ~ s . ~ ’ - ~ ~ Fasciitis without bony involvement is well-defined on magnetic

resonance imaging, whereas intramuscular lesions are poorly defined. Subcutaneous lesions are well-defined on CT48 but none of these imaging techniques are specific. CT was useful in this case as it was highly indicative of a benign lesion. Fasciitis has been diagnosed by aspiration cytol-

Local excision is usually curative, even if in- complete, although recurrence (or probably per- sistence) occurs in a few cases, usually within a few weeks but occasionally &r several m0nths?,16917~21 Fasciitis can be self-limiting and cases of sponta- neous resolution have been reported.4,15*25,50*51 This behavior is more suggestive of a reactive than neoplastic process, and flow cytometry has confirmed the diploid nature of f a ~ c i i t i s . ~ ~

Initiating factors are poorly understood. Trauma has been implicated but Meister et a1.18 found a convincing history in only 5% of cases. Many cases of head and neck fasciitis develop at sites susceptible to trauma, such as bony promi- nences at the mandibular angle, inferior border of the mandible, and anterior mandible, and zygo- matic arch.17 A viral origin and thrombotic mech- anisms have also been proposed.52

The gross appearance of resected specimens has been reported as highly ~ a r i a b l e . ’ . ~ , ~ ~ Most are round or oval and well-circumscribed, but not encapsulated, although some have poorly defined

ogy.49,50


margins. Consistency varies from firm to rubbery to soft, and color has been described as gray- white, reddish-pink, tan, or yellowish-white, The cut specimen may show a solid mass with a fibrous or gelatinous surface, or cystic areas.

Histologic interpretation of soft tissue tumors is difficult. Fasciitis can appear initially alarm- ing as i t lacks encapsulation and extends locally into adjacent tissue incorporating nerves vessels and small islands of muscle or fat. Histologic classification has been proposed but not universally a ~ c e p t e d . ' ~ , ~ ~ This is a reflection of diverse histologic appearances with often more than one histologic type present in a single specimen. Allen7 identified four features common to nearly all cases of fasciitis: (1) spindle-shaped fibroblasts that tend to be arranged in long fascicles which are slightly curved, whorled, or S-shaped; (2) small vascular clefts or slit-like spaces that often separate the fibroblasts; (3) extravasation of erythrocytes; and (4) mucoid interstitial ground substance. The variable histologic appearances may reflect local tissue age and activity.14 Ran- domly arranged spindle, plump, or stellate fibro- blast-like cells embedded in a myxomatous stroma with abundant small vascular spaces and erythrocyte extravasation are suggestive of growth and relative immaturity, and this has been confirmed by cytometric analysis.26 By con- trast, spindle-shaped cells and bundles of inter- lacing collagen with infrequent vascular struc- tures a r e more suggestive of quiescence. Frequently, the appearance is variable and some- where between these two extremes, but the pre- dictable natural history and failure to equate histologic appearance with different behavior mean that classification is a purely academic exercise. Actually making the diagnosis of fasciitis is more import ant .

This case exhibited many typical features of fasciitis with the bulk of the lesion composed of loosely arranged fascicles of spindle-shaped cells with vascular clefts, although little erythrocyte extravasation. Scattered giant cells are not unusual and occasionally resemble osteoclasts as they did in this c a ~ e . ~ . ~ ~ In addition, there was some reactive bone rimmed by plump osteoblasts with other areas of more metaplastic bone, which is recognized as occurring in periosteal and cranial f a ~ c i i t i s . ~ ~ Of fundamental importance in di- agnosing fasciitis is the absence of abnormal mitoses, although mitosis number can be high as in this case.7,14,15 Other histologic features which may occur but were not seen in this case include

a reas of focal necrosis and mucin-filled ~ y s t s . ~ ~ , ' ~ , ' ~ , ~ ~ Despite a good understanding of the histologic features of fasciitis, differentiation from fibrous histiocytoma can be extremely difficult. 1,47

Ultrastructurally, the spindle or ganglion-like cells show features consistent with fibroblasts and m y o f i b r o b l a ~ t s . ~ ~ * ~ ~ , ~ ~ Im munohistochemical studies support this and also indicate a minor his- tiocytic element.2'47 A diagnosis of fasciitis is based largely on light microscopy, although im- munohistochemistry may be used in differentiat- ing fasciitis from other lesions, such as fibromatosis, fibrous histiocytoma, neurofibroma, and f ibrosar~oma.~~'~ ' However, immunohistochemis- try and electron microscopy are unable to differ- entiate between benign and malignant lesions.'

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Periosteal fasciitis of the mandible