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Revista Española de Cirugía Oral y Maxilofacial

On-line version ISSN 2173-9161Print version ISSN 1130-0558

Rev Esp Cirug Oral y Maxilofac vol.29 n.1 Barcelona Jan./Feb. 2007

 

CASO CLÍNICO

 

Nodular fascitis of the maxillofacial region. Two case reports and a review of the literature

Fascitis nodular en el territorio maxilofacial. Presentación de dos casos y revisión de la literatura

 

 

F. Almeida1, M. Picón2, M. Pezzi1, E. Sánchez-Jaúregui1, R. Carrillo3, J.L. Martínez-Lage4

1 Médico Residente.
2 Médico Adjunto.
3 Médico Adjunto. Servicio de Anatomía Patológica.
4 Jefe de Servicio. Servicio de Cirugía Oral y Maxilofacial.
Hospital Universitario Ramón y Cajal. Madrid, España

Dirección para correspondencia

 

 


ABSTRACT

Nodular fasciitis is a relatively rare clinical entity that is characterized by a rapidly growing tumor-like mass, although it has a benign clinical behavior and pseudosarcomatous histological characteristics. Its appearance in the maxillofacial area is unusual, and two cases are presented of patients that attended as a result of nodules that were hard in consistency, non-tender, in the zygomatic region beside the mandible, and that were diagnosed as nodular fasciitis. The cases are highlighted because of the atypical location of the nodular fasciitis and the histopathology is revised.

Key words: Nodular fasciitis; Pseudosarcomatous fibromatosis; Infiltrative fasciitis.


RESUMEN

La fascitis nodular es una entidad clínica relativamente poco frecuente caracterizada por una tumoración de crecimiento rápido, aunque de comportamiento clínico benigno y de características histológicas pseudosarcomatosas. Su aparición en el territorio maxilofacial es poco común, por lo que presentamos dos casos que acudieron por nódulos de consistencia dura, indoloros, en las regiones cigomática y paramandibular, que fueron diagnosticados de fascitis nodular. Destacamos estos casos por tratarse de fascitis nodular de localización atípica y revisamos su histopatología.

Palabras clave: Fascitis nodular; Fibromatosis pseudosarcomatosa; Fascitis infiltrativa.


 

Introduction

Nodular fasciitis was described originally by Konwaler and cols in 1955 under term subcutaneous pseudosarcomatous fibromatosis (fasciitis).1 It is a clinical entity that is not very common, in spite of its pseudosarcomatous histological characteristics, with behavior that is clinically benign. In 1966 Mehrengan and cols described 17 cases with epidemiological, clinical and histopathological characteristics, and they proposed the term nodular fasciitis because it was briefer and more descriptive.2 Although it is a mesenchymal lesion commonly found in soft tissue in other locations, especially in the upper limbs and trunk, its appearance in the head and neck is not very common, 3 and the oral and maxillofacial surgeon may not be very familiar with it.

Two cases are presented of nodular fasciitis that were seen by the Service of Oral and Maxillofacial Surgery of the Hospital Ramón y Cajal (Madrid) that arose in the subcutaneous tissue in the zygomatic region beside the mandible.

 

Case reports

Case 1

The patient was a 39-year-old female, non-smoker, with no known medical allergies and with no medical-surgical history of interest. She attended the outpatient unit because of a non-tender, tumor-like mass that had been growing rapidly in the right zygomatic area over the previous two months. The clinical examination showed a nodule that measured approximately 1.5 cm in greatest diameter, that was hard in consistency, non-tender and that affected the subcutaneous cell tissue. It was not adhered to the malar bone. No adenopathy in the neck was noted. MRI of the face was carried out, and a small solid nodule was reported that was isointense on T1 and T2 after contrast enhancement. It had a superficial location in the region of the right malar (Fig. 1). Following the fat suppression sequence, lipoma was excluded. The report was of a soft tissue tumor (fibroma, leiomyoma, sarcoma, etc.) with a 13 mm diameter, and FNA was carried out, which gave the result of mesenchymal proliferative lesion, that was suggestive of nodular fasciitis. Surgical excision of the nodule was carried out intraorally. The histological study of the extracted nodule revealed a proliferation of spindle-shaped cells that was moderately dense, and distributed in haphazardly arranged bundles in a "feather shape". There were large vesicular nuclei and moderate mitotic activity. Between the cells a loose or myxoid collagenous matrix could be observed with some inflammatory cells and extravasated red blood cells (Fig. 2). The borders of the lesion were distinctly irregular. The anatomopathological diagnosis was of nodular fasciitis. The patient is currently asymptomatic after a follow-up of 24 months.

Case 2

The patient was a 21-yearold female, non-smoker, with no medical-surgical history of interest, and no known drug allergy. She presented with a subcutaneous tumorlike mass that was attached to the skin, on the right edge of the face by the mental crease with central cutaneous invagination. It had been evolving for three months and it was growing progressively (Fig. 3). There was no history of trauma in the area. On examination a nodule could be felt with a maximum diameter of 2 cm. It was hard in consistency, slightly painful, and it was attached to the skin. It was affecting subcutaneous cellular tissue and there was no involvement of the mandibular bone. The CAT scan showed a nodular lesion that was located in the subcutaneous tissue in the right mandibular region, which had a diameter of 2 cm There was no bone erosion, nor was there extension to adjacent layers. FNA was carried out of the nodule and a mesenchymal proliferative lesion was reported, suggestive of a reactive process of a nodular fasciitis type.

With the presumed diagnosis of nodular fasciitis, extraoral surgical excision was carried out of the node. The histological study of the excised lesion showed a proliferation of spindle-shaped cells that appeared to be reactive, very similar to those observed in Case 1. The lesion was also diagnosed as nodular fasciitis. The patient is currently asymptomatic after a follow-up of 14 months.

 

Discussion

Benign fibroblastic proliferation represents a group of heterogeneous entities that are well-defined, and that are reactive rather than true neoplasms. Nodular fasciitis is found among theses entities together with proliferative fasciitis and proliferative myositis. All have rapid growth and rich cellularity.4

Nodular fasciitis has throughout history been given different names, such as infiltrative fasciitis, pseudosarcomatous fasciitis and pseudosarcomatous subcutaneous fibromatosis. It is a lesion that affects patients of all ages that is very rarely described, although the incidence rate is higher in the third and fifth decades in life. Significant differences in male and female prevalence have not been appreciated. It can appear in all areas of the body except the viscera, although the most common location is in the upper limbs and torso. 7-20% of cases are located in the maxillofacial area, especially in the subcutaneous tissue of the malar and/or mandibular region, as in the cases presented. Intraoral lesions are exceptional.5

It is a benign proliferative lesion of myofibroblasts that commonly appears in subcutaneous tissue or between muscle fasciae, although on rare occasions it will arise within the muscle tissue thickness. Its etiology is unknown, but it is thought that repeated trauma to the affected area could influence its pathogenesis, as it commonly arises in prominent areas such as the mandibular angle and body, and the zygoma.6 Selflimiting cases have been described with spontaneous remission of the nodule, which would support this pathogenic hypothesis. It appears as a rapidly growing lesion of the dermis or submucosa. The lesion is hard and well-circumscribed, although not encapsulated, and it is generally non-tender.

Clinical behavior in the area of the head and neck is similar to that in other locations and it is characterized by rapid growth of the mass over weeks, although Price and cols. affirm that in most cases the nodule grows slowly or even not at all.7, 8 In our cases one of them had rapid growth (Case 2) while in the other growth was slow or not at all. The lesions were nodular, hard and non-tender.

From the histological point of view, nodular fasciitis is considered a reactive proliferation, more than tumoral, of cells with myofibroblastic differentiation.9 When it was first described, its histological similarity with the soft tissue sarcoma was highlighted, leading to terms for it that included "pseudosarcomatous". Over time, the lesion has become well-characterized and its histological diagnosis does not tend to give the experienced pathologist any problems. The presence of large spindle-shaped cells, but without any true pleomorphism and nuclear atypia is very constant. In addition this cellularity is arranged in loose bundles, haphazardly arranged, in a so-called "feather" shape that is reminiscent of cell culture. There may be abundant mitoses but no atypical figures are shown. Foci of inflammatory infiltrate and extravasated red blood cells are also very characteristic. Only if small, incomplete samples are received, or samples with artifacts, will problems arise in its histologic diagnosis. The clinico-pathologic correlation is, in these cases, very important. Its superficial location in subcutaneous tissue or fascia, its small size and rapid growth are all very characteristic details.

Surgical resection is the treatment of choice. Good results are obtained even in cases where the excision is incomplete.10 Intralesional corticoid therapy with injections has been proposed, but this should be reserved for very specific cases.11 If there is recurrence, incomplete resection should be suspected, or the original diagnosis should be revised.10

 

 

Dirección para correspondencia:
Fernando Almeida Parra
C/ Cardenal Marcelo Spinola 48, 17ºC
28016 Madrid, España
E-mail: falmeida.hrc@salud.madrid.org

Recibido: 24.10.05
Aceptado: 06.10.06

 

References

1. Konwaler BE, Keasbey L, Kaplan L. Subcutaneous pseudosarcomatous fibromatosis (fascitis). Am J Clin Pathol 1955;25:241-52.        [ Links ]

2. Mehregan AH. Nodular fascitis. Arch Dermatol 1966;93:204-210.        [ Links ]

3. Bernstein KE, Lattes R. Nodular (pseudosarcomatous) fascitis, a non-recurrent lesion. Clinicopathological study of 134 cases. Cancer 1982;49:1668-78.        [ Links ]

4. Mauleón C, Chavarría E, Suárez R et al. Fascitis Nodular. Actas Dermosifiliogr 2003;94:90-2.        [ Links ]

5. Martínez-Blanco M, Bagán JV, Alba JR, Basterra J. Maxillofacial nodular fascitis: a report of 3 cases. J Oral Maxillofac Surg 2002;60:1211-4.        [ Links ]

6. DiNardo LJ, Wetmore RF, Potsic WP. Nodular fasciitis of the head and neck in children. A deceptive lesion. Arch Otolaryngol Head Neck Surg 1991;117:1001-2.        [ Links ]

7. Price EB, Silliphant WM, Shuman R. Nodular fasciitis: a clinicopathologic analysis of 65 cases. Am J Clin Pathol 1961;35:122-36.        [ Links ]

8. Nair P, Barrett AW, Theodossy T. Oral nodular fascitis: case report. Br J Oral Maxillofac Surg 2004; 42:360-2.        [ Links ]

9. Dayan D, Nasrayah V, Vered M. Clinico-pathologic correlations of myofibroblastic tumors of the oral cavity: 1. Nodular fasciitis. J Oral Pathol Med 2005; 34: 426-35. Review.        [ Links ]

10. Silva P, Bruce IA, Malik T, Homer J, Banerjee S. Nodular fasciitis of the head and neck. J Laryngol Otol 2005;119:8-11.        [ Links ]

11. Grabam B, Barret T, Goltz R. Nodular fascitis: responde to intralesional corticoesteroids. J Am Acad Dermatol 1999;40:490-92.        [ Links ]

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