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Incidence and treatment of recurrent plantar fibromatosis by surgery and postoperative radiotherapy
The American Journal of Surgery 187 (2004) 33–38
Scientific paper
Incidence and treatment of recurrent plantar fibromatosis by surgery and postoperative radiotherapy Eelco de Bree, M.D.a, Frans A. N. Zoetmulder, M.D., Ph.D.a,*, Ronald B. Keus, M.D.b, Hans L. Peterse, M.D.c, Frits van Coevorden, M.D, Ph.D.a a
Department of Surgical Oncology, The Netherlands Cancer Institute/Antoni van Leeuwenhoek Huis, Plesmanlaan 121, 1066 CX Amsterdam, Netherlands b Department of Radiotherapy, The Netherlands Cancer Institute/Antoni van Leeuwenhoek Huis, Amsterdam, Netherlands c Department of Pathology, The Netherlands Cancer Institute/Antoni van Leeuwenhoek Huis, Amsterdam, Netherlands Manuscript received April 29, 2002; revised manuscript November 16, 2002
Abstract Background: The incidence of plantar fibromatosis (PF) is unknown. Sometimes PF tends to recur repetitively after surgical treatment. In our institute we have used postoperative radiotherapy in an attempt to diminish the change on recurrence. Methods: The Dutch Network and National Database for Pathology (PALGA) was consulted to establish the incidence of plantar fibromatosis (PF). Data from 9 patients (11 feet) with PF referred to our institute for recurrent disease were analyzed and the role of postoperative radiotherapy in prevention of recurrence was studied. Results: An average of 1.2 operations for PF was performed per 100,000 citizens yearly in the Netherlands. Twenty-six operations were performed and postoperative radiotherapy was used in 6 cases. Plantar fasciectomy was associated with the lowest recurrence rate. After microscopically incomplete excision or excision of early recurrence (ⱕ6 months) alone all tumors recurred, while recurrence was rarely observed after adjuvant radiotherapy. However, radiotherapy was associated with significantly impaired functional outcome in 3 cases. Conclusions: Plantar fibromatosis is relatively rare. Plantar fasciectomy seems to be the operation of choice. Although effective in decreasing the recurrence rate, adjuvant radiotherapy should be used very selectively because of its serious side effects. © 2004 Excerpta Medica, Inc. All rights reserved. Keywords: Plantar fibromatosis; Incidence; Treatment; Surgery; Radiotherapy
Plantar fibromatosis (PF) is a proliferation of fibroblasts involving the plantar aponeurosis. Fibromatosis is usually localized at sites sensitive to stress, including hand palm, foot, arm, leg, neck, shoulder girdle and abdominal wall. Therefore, it remains unclear whether PF has to be seen as an overreaction to trauma or as a neoplasm. It tends to have a benign clinical behavior with an insidious onset and slowly progressive course; eventually it is locally aggressive with invasion of the overlying skin or deep structures, but metastases do not occur. Considering its clinical behavior, PF has to be seen as a separate entity from aggressive fibromatosis of the extremities [1]. Plantar fibromatosis has usually a milder biological
* Corresponding author. Tel.: ⫹0031-20-5122550; fax: ⫹0031-205122554. E-mail address: [email protected]
behavior. While aggressive fibromatosis tends to recur as skip lesions, each recurrence more proximally, eventual recurrence of PF is usually observed more locally. Generally, PF responds initially to nonoperative measures such as orthoses or corticosteroid injection. Surgery is indicated only if the lesions are highly symptomatic, because surgical treatment is associated with high recurrence rates and significant morbidity [1,2]. We report on the experience with the management of PF at our institute during the last decades. As our hospital is a referral hospital, most cases presented at our hospital with recurrent disease. Analogous to the management of fibromatosis of trunk and extremities [1– 6], adjuvant radiotherapy was given in a number of our cases. The additional use of radiotherapy in PF is mentioned only sporadically in the literature [1,2]. The therapeutic benefit and complications of this approach are discussed. Additionally an effort was made to establish the incidence of operations for PF.
0002-9610/04/$ – see front matter © 2004 Excerpta Medica, Inc. All rights reserved. doi:10.1016/j.amjsurg.2002.11.002
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E. de Bree et al. / The American Journal of Surgery 187 (2004) 33–38
Patients and methods The Dutch Network and National Database for Pathology (PALGA), including the histological reports from all Dutch pathology departments, supplied information on the incidence of PF in the Netherlands during the period from January 1, 1995, until December 31, 1999. Only total numbers of histological diagnosis of PF could be identified, without differentiating between primary and recurrent disease. A search in the records of our institute over a 20-year period identified 9 patients with PF in 11 feet. The clinical diagnosis was confirmed by histological examination in all but 1 case. In 1 case clinical, radiological, and cytological findings revealed the diagnosis of PF. Not only treatment data from our hospital were gathered, but also data from previous treatment in other hospitals were analyzed retrospectively. Factors identified in the records included age at diagnosis, sex, side of location, eventual period of conservative treatment in our hospital, type and time of primary and secondary surgical treatment, use of adjuvant radiotherapy, treatment complications, and follow-up for recurrence and subjective foot function, starting from time of diagnosis. Additionally, risk factors for development of PF (see discussion) were noted. All operation and histological reports concerning treatment in our hospital as well as in other hospitals were gathered and analyzed. Our pathologists in all cases revised histology from other hospitals. The surgical procedures performed include marginal excision, wide local excision (with at least 1 cm of surrounding macroscopic normal tissue) and subtotal fasciectomy. Excision of the overlying skin and the use of full or split skin grafting were noted. Resections were categorized as macroscopically incomplete, microscopically incomplete, and microscopically complete.
Results Incidence of surgery for plantar fibromatosis in the Netherlands In the 5-year period mentioned, surgical specimens with PF were offered for histological examination 173 to 194 times (average 182) per year in the Netherlands (Table 1). In a country with approximately 15 million citizens, this means an average of 1.2 operations for PF per 100,000 citizens yearly. The age and sex distribution is demonstrated in Table 1, and there were no significant differences in between the consecutive 5 years. Characteristics of patients treated The records of 9 patients, 6 female and 3 male, were studied. Eleven feet, 8 left and 3 right, had PF. The age of the patients at diagnosis varied from 8 to 46 years, with a
Table 1 Age and sex distribution of patients with plantar fibromatosis from whom histological material was obtained in the Netherlands from January 1, 1995, until December 31, 1999 Age (years)
Total
Male
Female
0–9 10–19 20–29 30–39 40–49 50–59 60–69 70–79 80⫹ Total
11 (1%) 26 (3%) 99 (11%) 207 (23%) 239 (26%) 224 (25%) 76 (8%) 27 (3%) 2 (0%) 911 (100%)
6 (1%) 15 (3%) 68 (14%) 153 (31%) 126 (26%) 87 (10%) 26 (5%) 10 (2%) 2 (0%) 493 (100%)
5 (1%) 11 (3%) 31 (7%) 54 (13%) 113 (27%) 137 (33%) 50 (12%) 17 (4%) 0 (0%) 418 (100%)
median and mean age of 25 and 26 years, respectively. Eight patients were referred to our hospital for treatment of recurrent disease (5 feet) or for adjuvant radiotherapy after surgery for local recurrence elsewhere (3 feet). Two patients treated for recurrent disease developed PF at the opposite foot and were subsequently treated again. One patient presented during follow-up examination for treated breast cancer in situ with a nodule of 1.5 cm at the foot sole. Clinical findings and ultrasonography suggested PF. Fine-needle aspiration revealed fibroblastic proliferation, leading to the diagnosis PF. Risk factors Two patients developed bilateral PF, and a 33-year-old patient had a history of multiple excision of a recurrent tumor on her other foot sole when she was 8 years of age. According to this history it might have been also plantar fibromatosis, but we were not able to trace histological data. The same patient had bilateral palmar fibromatosis and a mother with unilateral PF. Another patient developed unilateral palmar fibromatosis during the follow-up period and the father of her mother had been diagnosed with bilateral palmar fibromatosis. Two patients developed PF a few years after local penetrating trauma (needle and metal nail). Primary treatment At diagnosis, all but 3 patients had symptomatic disease. After initial increase in size from 1.5 to 2.5 cm in diameter, spontaneous regression occurred during the next 2 years in 1 patient. At present a lesion of only a few millimeters in size has remained. Two other patients with initially asymptomatic PF were left untreated for periods of 9 months and 3.5 years, until substantial symptoms arose. Absence of response to nonsurgical means necessitated surgical treatment. A total of 26 operations were performed for these 11 sites of PF (Table 2). For primary PF marginal excision was performed in 6 patients, wide local excision in 3 cases, and
E. de Bree et al. / The American Journal of Surgery 187 (2004) 33–38 Table 2 Frequency of recurrence after surgery only and surgery with postoperative radiotherapy
Microscopical completeness of excision Microscopically incomplete Microscopically complete Time to any recurrence and outcome of subsequent treatment of recurrence ⱕ6 months ⬎6 months Surgery for primary plantar fibromatosis Marginal excision Wide local excision Plantar fasciectomy Surgery for recurrent plantar fibromatosis Marginal excision Wide local excision Plantar fasciectomy Below-knee amputation
Surgery only
Surgery and radiotherapy
3/3 12/16
1/4 0/2
3/3 3/6
1/5 0/1
6/6 2/3 1/1
— — —
3/3 2/3 0/1 1/2
0/2 1/3 0/1 —
incompleteness of previous excision necessitated a belowknee amputation. In conclusion, plantar fasciectomy for primary and recurrent PF was associated with the lowest recurrence rate (33%) compared with marginal and wide local excision (82% and 56%, respectively). Value of skin excision Overlying skin was excised in 8 surgical procedures; in only 1 case the skin was microscopically involved. Excluding the last case, because the operation was converted to a below-knee amputation a couple of weeks later, tumor recurred in 4 of the 7 cases after excision of the tumor including overlying skin. A skin transplantation had to be performed to close the defect in 5 cases. Split- and fullthickness skin graft were used each in 2 cases, while in 1 report the type of skin transplant was not mentioned. Adjuvant radiotherapy
plantar fasciectomy in 1 case. Although in only 1 case histological examination demonstrated incomplete local excision of the primary lesion, in 9 primarily surgically treated feet tumor recurred, resulting in a recurrence percentage of (90%). First recurrence was observed 1 to 27 months (mean 9, median 10) after primary surgery. In 1 case asymptomatic recurrence is left untreated until time of this investigation. All other recurrences were operated on. Re-recurrence and its treatment Tumor recurred for the second time in 4 of 8 feet treated surgically for recurrent disease (50%). Treatment and outcome are summarized in Table 3. Marginal excision was performed 5 times for recurrent PF, wide local excision in 6 cases, and plantar fasciectomy in 2 patients. In 1 patient recurrence above the ankle made below-knee amputation necessary. Subsequently, this patient presented with a third recurrence at her knee. After local excision, tumor recurred at the same site for the fourth time and was successfully treated by local excision and radiotherapy. In another case a third recurrence appeared. The local recurrence was treated by microscopically incomplete wide local excision of tumor and overlying skin. In this case, impairment of foot function after previous radiotherapy, wound healing problems, and Table 3 Severe side effects of postoperative radiotherapy in 6 cases Dystrophic foot with impaired foot function Femur fracture†
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3 cases* 1 case
* In 1 case combination with wound healing problems after microscopically incomplete excision of repeated recurrence necessitated below-knee amputation. † Fourteen years after postoperative radiotherapy for recurrence at knee level.
Adjuvant radiotherapy was applied during the treatment of 6 localizations of PF. In 3 cases 40 Gy was applied to the foot in a generous field reaching to above the ankle with an additional boost of 20 Gy to the foot sole. In 2 cases 60 Gy was given only to the foot sole, while in 1 case of recurrence at knee level 60 Gy was administered to the entire leg remaining after below-knee amputation. In 4 cases it was used after microscopically incomplete excision of recurrent PF, and in another 2 cases as adjuvant treatment after surgery for very early recurrence of PF, 1 and 4 months after initial excision. The results of adjuvant radiotherapy, compared with outcome after surgery only are summarized in Table 2, while the side effects of postoperative radiotherapy are noted in Table 3. Follow-up and functional outcome The mean and median follow-up periods, counted after initial diagnosis of each foot with PF, are 17 and 20 years (range 2 to 26). All patients treated surgically are alive and all but 1 free of disease for the last 13 to 24 years (mean 20 and median 19 months). One patient developed asymptomatic recurrence 2 years ago and is left untreated until today. Two treated feet became dystrophic after surgery and adjuvant radiotherapy and these patients are unable to walk without pain, seriously impairing their quality of life. Two patients are able to walk relatively satisfactory with leg prosthesis after below-knee amputation. The 5 remaining patients retained good leg and foot function.
Comments The exact etiology of PF is unknown. It remains unclear whether PF has to be seen as an overreaction to trauma or as a real neoplasm. A hereditary role has been suggested as
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familial occurrence of plantar and palmar fibromatosis has been reported [7]. Other risk factors reported include history of contralateral plantar fibromatosis, palmar fibromatosis (Dupuytren’s disease) and penile fibromatosis (Peyronie’s disease), trauma, infection, neuropathy, epilepsy, gout, tuberculosis, and chronic intoxication (chiefly lead poisoning) [8 –10]. In the reported series bilateral involvement is observed in a widely varying frequency from 10% to 67% of the patients [1,9 –15]. A wide variation in age at onset exists. In reported series the mean age of treated patients varied from 32 to 49 years [9,12–17]. Our population was significantly younger, but PF has been seen to occur also in childhood [11]. In our Dutch Network and National Database for Pathology, patients operated for PF were most frequently in the fourth to sixth decade of life. Only 4% of patients were younger than 20 years of age and a similar percentage of patients older than 70 years of age. Reports on sex predominance have been contradictory [9 –17]. In our histological archive survey the male-female ratio was 1.2:1. Remarkable is that the highest incidence of histological diagnosis of PF was noted a decade later in females than in males (Table 1). The left foot seems to be slightly more often affected [9 –17]. Plantar fibromatosis generally starts as an asymptomatic nodule in the plantar fascia. Symptoms include pain and swelling on the sole of the affected foot. Despite numerous reports on palmar fibromatosis contractures, similar plantar lesions are rarely observed [18]. Diagnosis is mainly established after physical examination. In an active and early stage, the tumor nodules in tends to be less well defined, softer, and not adherent to the overlying skin, while in a late or resting stage, the lesions is more discrete, harder, commonly adhered to the overlying skin, and measures more than 1 cm. The differential diagnosis includes many malignant and benign soft tissue tumors, including fibrosarcoma and other sarcomas, lipoma, giant cell tumor of tendon sheath, neurofibroma, neurilemmoma, granuloma annulare, rheumatoid nodule, ganglion, epidermoid inclusion cyst, sweat gland carcinomas, and intradermal melanoma [19]. Ultrasonography, computed tomography, and magnetic resonance imaging may visualize size, shape, invasiveness, and exact location [20 –22]. Magnetic resonance imaging is at present considered the modality of choice in the assessment of PF. At histological examination, active proliferation features of fibroblastic cells with many large and hyperchromatic nuclei, loose interstitial collagen fibers, and a mild degree of cellular atypia may be seen. In contrast, in its resting or latent state, large amount of thick and mature fibrous tissue with low cellularity and complete absence of cellular pleomorphism are the common histological features [19,23]. Immunohistochemical examination and ultrastructural analysis reveal similarities with Dupuytren’s disease [24]. The natural history of PF remains one of unpredictable, usually slow, and locally invasive growth. When PF is considered as local fibroblastic proliferation in reaction on
chronic trauma to the plantar fascia, including incorporation of a corpus alienum and insufficient pressure distribution during walking, it is understandable that in most of these cases the lesion remains asymptomatic and stable in size for prolonged periods of time and that spontaneous regression and disappearance occurs [11]. The surgeon is usually consulted for persisting and symptomatic disease. Our survey suggests that approximately one operation for primary or recurrent PF is performed per 100,000 citizens yearly. In other words, a Dutch surgeon operates on PF an average of once in 6 to 7 years. When the lesion recurs after excision, its natural behavior is usually less mild and a more aggressive treatment is warranted. Most of the patients in our series and in other reports belong to the latter group. Nonoperative management is the mainstay of treatment because the disease is often asymptomatic or only mildly symptomatic and responsive to measures such as antiinflammatory medication, modification of activities, physical therapy, orthoses, or occasionally corticosteroid injection [8,10 –15,17,18,23,25,26]. Surgery is indicated only if the lesions are highly symptomatic, locally aggressive, attempts at nonoperative management fail, or if the diagnosis is in question. Surgical treatment is associated with high recurrence rates and significant morbidity [8,10 –14,17,18,23,25]. Although leaving residual disease behind is one of the risk factors for fibromatosis recurrence, microscopically incomplete excision is not automatically followed by local recurrence [2,26,27]. However, in our small series all microscopically incomplete excised tumors recurred, unless adjuvant radiotherapy was administered. Other factors identified with an increased risk for recurrence were multiple nodules, bilateral lesions, and positive family history [12]. When presenting at a young age (ⱕ18 to 30 years), there seems to be a greater likelihood of recurrence after surgery (P ⫽ 0.007 to 0.056) [1,12]. Others support a more favorable course in children [11]. The surgical procedures performed for PF include marginal excision, wide local excision (with a minimal margin of 1 to 2 cm surrounding normal tissue), and subtotal plantar fasciectomy with or without skin grafting, full or split thickness). Different operation techniques have been described for plantar fasciectomy, especially regarding design of the skin incision [8,9,13,15,25]. After local excision of the primary tumor recurrence rates varying from 40% to 100% are reported in different series with probably a negative selection of patients, as discussed above [9,10,12,17]. The high recurrence rate after marginal excision can be explained by the diffuse nature of the lesion along the plantar fascia. In these series, wide local excision and subtotal fasciectomy were more successful as primary treatment, with recurrence rates of 20% to 33% and 0% to 50% respectively [12–17]. Marginal excision as treatment of recurrent disease was associated with a recurrence rate of 70% to 100%, comparable with our results [9,10,12,14]. While others observed an equal failure rate of 75% to 78% after wide local excision of recurrent disease [12,14], Wap-
E. de Bree et al. / The American Journal of Surgery 187 (2004) 33–38
ner et al [13] reported no recurrence after 7 such excisions. It has to be noted that they performed such a wide local excision that this procedure might be called also subtotal plantar fasciectomy in some cases. As also observed in our series, plantar fasciectomy seems to be the most successful procedure and is associated with a failure rate of 0% to 38% [9,12,14]. When PF is seen as local reactive fibrotic tissue due to chronic trauma to the plantar fascia and not as tumor, complete excision of the plantar fascia seems to be logically also the most optimal procedure. Local excision may lead to new fibrotic scar tissue and permits new chronic trauma to the fascia, resulting in high risk on recurrent disease. Complications have been observed up to 18% and 31% of cases after surgery for primary and recurrent PF respectively and include mainly wound healing problems, chronic pain and poor functional outcome [12,13,15]. In only a few studies functional outcome was noted and the results were judged functionally satisfactory in 70% to 80% of cases [12,13,15] In our series functional outcome was good in 60% of treated feet. In the literature, below-knee amputation had to be performed in a widely varying percentage of cases (0% to 22%) for misdiagnosis of fibrosarcoma, more proximal recurrences of aggressive fibromatosis, wound healing problems, severe impaired function of the foot and intractable pain [1,12,13,15]. In 1 of our cases wound healing problems and functional impairment after surgery and radiotherapy necessitated amputation. In another case recurrence to above the ankle made amputation inevitable, but not preventing another recurrence at the knee level. It may be discussed whether this case was more likely to be typical aggressive fibromatosis from the onset rather than PF. Excision of uninvolved overlying skin does not decrease recurrence rate significantly. Involved overlying skin however should be adequately excised and may result in a skin defect, necessitating skin grafting. There is no difference in results or complications between full- and split-thickness skin grafting. Mean time to weightbearing on the affected feet after skin grafting is only slightly longer [12]. Recently, autogenous free deepithelialized dermal fat grafting after primary excision has been proposed as a means to fill up the defect preserving a soft, supple, weightbearing surface with minimal scar tissue formation [28]. Interposition of Marlex surgical mesh after excision of plantar fascia has been advocated for functional restore, but only a small subjective improvement was observed [16]. The use of radiotherapy has been only sporadically reported in the management of PF and therefore its role remains unclear [1,2,9]. Spear et al [1] reported local control of the disease in 2 patients treated by combination of surgery and radiotherapy. Primary radiotherapy as only treatment for recurrent tumor has not been curative in 5 cases reported [1,2,9]. In the treatment of primary and recurrent fibromatosis at other sites, postoperative radiotherapy may result in an increased local control rate [1–5]. While wide local excision with negative margins is the treatment of choice for aggressive fibromatosis, function-sparing resec-
37
tion is appropriate because adjuvant radiotherapy can offset the adverse impact of positive margins [1,4,5]. In our small series, adjuvant radiotherapy was associated with decreased risk for recurrence. After marginal excision, lower extremities remained only free of disease when adjuvant radiotherapy was added. Also after wide local excision radiotherapy resulted in a 50% reduction of the recurrence rate. While all tumors recurred after microscopically incomplete excision only, only 1 of 4 tumors with positive surgical margins recurred when radiotherapy was added postoperatively. When time to recurrence is 6 months or less, subsequent surgery only resulted in recurrence in all cases, while in only 1 of 5 cases recurrence was observed after adjuvant radiotherapy. Radiotherapy doses of 50 to 65 Gy seem to be effective as adjuvant treatment [1– 6,27]. Although adjuvant radiotherapy may result in improved local control, radiation induced impaired function of the foot, lymph edema, marked fibrosis and fracture of irradiated bone might be the price to be paid for this therapeutic benefit [5,29]. Impaired foot function and wound healing after radiotherapy may even lead to amputative surgery. In our series, radiotherapy contributed significantly to the need for amputation in 1 case and resulted in invalidating foot dystrophy in 2 cases. Additionally, the risk for radiation-induced malignancy exists after radiotherapy for this benign disease. In conclusion, marginal local excision regularly leads to local relapse. Plantar fasciectomy is the treatment of choice for primary or recurrent symptomatic PF. Excision of overlying uninvolved skin does not decrease the recurrence rate. Wide excision of extensive disease may result in significant morbidity as a result of excision or damage to adjacent anatomical structures. This implies the need for more local function and structure-preserving procedures in these cases, despite the association with a high recurrence rate. In case of microscopically incomplete surgery or surgery for very early recurrence of PF, adjuvant radiotherapy may diminish the risk on recurrence. However, postoperative radiotherapy of the foot is associated with a high rate of severe adverse effects. In view of its potentially serious side effects, postoperative radiotherapy should be used highly selectively for this benign disease. The risk of complications needs to be weighed against the risk of observation alone. References [1] Spear MA, Jennings LC, Mankin HJ, et al. Individualizing management of aggressive fibromatosis. Int J Radiat Biol Phys 1998;40:637– 45. [2] Miralbell R, Suit H, Mankin HJ, et al. Fibromatoses: from postsurgical surveillance to combined surgery and radiation therapy. Int J Radiat Oncol Biol Phys 1990;18:535– 40. [3] Keus R, Bartelink H. The role of radiotherapy in the treatment of desmoid tumors. Radiother Oncol 1986;7:1–5. [4] Ballo MT, Zagars GK, Pollack A, et al. Desmoid tumor: prognostic factors and outcome after surgery, radiation therapy, or combined surgery and radiation therapy. J Clin Oncol 1999;17:158 – 67.
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[5] Nuyttens JJ, Rust PF, Thomas CR, Turrisi AT. Surgery versus radiation therapy for patients with aggressive fibromatosis or desmoid tumors: a comparative review of 22 articles. Cancer 2000;88:1517– 23. [6] Merchant NB, Lewis JJ, Woodruff JM, et al. Extremity and trunk desmoid tumors: a multifactorial analysis of outcome. Cancer 1999; 86:2045–52. [7] Chen KT, Van Duyne TA. Familial plantar fibromatosis. J Surg Oncol 1985;29:240 –1. [8] Landers PA, Yu GV, White JM, Farrer AK. Recurrent plantar fibromatosis. J Foot Ankle Surg 1993;32:85–93. [9] Pickren JW, Smith AG, Stevenson TW, Stout AP. Fibromatosis of the plantar fascia. Cancer 1951;4:846 –56. [10] Allen RA, Woolner LB, Chormly RK. Soft tissue tumors of the sole with special reference to plantar fibromatosis. J Bone Joint Surg 1955;37A:14 – 6. [11] Godette GA, O’Sullivan M, Menelaus MB. Plantar fibromatosis of the heel in children: a report of 14 cases. J Pediatr Orthop 1997;17: 16 –7. [12] Aluisio FV, Mair SD, Hall RL. Plantar fibromatosis: treatment of primary and recurrent lesions and factors associated with recurrence. Foot Ankle Int 1996;17:672– 8. [13] Wapner KL, Ververeli PA, Moore JH, et al. Plantar fibromatosis: a review of primary and recurrent surgical treatment. Foot Ankle Int 1995;16:548 –51. [14] Durr HR, Krodel A, Trouillier H, et al. Fibromatosis of the plantar fascia: diagnosis and indication for surgical treatment. Foot Ankle Int 1999;20:13–7. [15] Sammarco GJ, Mangone PG. Classification and treatment of plantar fibromatosis. Foot Ankle Int 2000;21:563–9. [16] Oster JA, Miller AE. Resection of plantar fibromatosis with interposition of Marlex surgical mesh. J Foot Surg 1986;25:217–25.
[17] Aviles E, Arlen M, Miller T. Plantar fibromatosis. Surgery 1971;69: 117–20. [18] Clasen DA, Hurst LN. Plantar fibromatosis and bilateral flexion contractures: a review of the literature. Ann Plast Surg 1992;28: 475– 8. [19] Wu KK. Plantar fibromatosis of the foot. J Foot Ankle Surg 1994; 33:98 –101. [20] Bedi DG, Davidson DM. Plantar fibromatosis: most common sonographic appearance and variations. J Clin Ultrasound 2001;29:499 – 505. [21] Recht MP, Donley BG. Magnetic resonance imaging of the foot and ankle. J Am Acad Orthop Surg 2001;9:187–99. [22] Robbin MR, Murphey MD, Temple HT, et al. Imaging of musculoskeletal fibromatosis. RadioGraphics 2001;21:585– 600. [23] Lee TH, Wapner KL, Hecht PJ. Plantar fibromatosis. J Bone Joint Surg 1993;75A:1080 – 4. [24] de Palma L, Santucci A, Gigante A, et al. Plantar fibromatosis: An immunohistochemical and ultrastructural study. Foot Ankle Int 1999; 20:253–7. [25] Curtin JW. Fibromatosis of the plantar fascia: surgical technique and design of skin incision. J Bone Joint Surg 1965;47A:1605– 8. [26] Pentland AP, Anderson TF. Plantar fibromatosis responds to intralesional steroids. J Am Acad Dermatol 1985;12:212– 4. [27] Lewis JJ, Boland PJ, Leung HY, et al. The enigma of desmoid tumors. Ann Surg 1999;229:866 –72. [28] Lauf E, Freedman BM, Steinberg JS. Autogenous free dermal fat grafts in the surgical approach to plantar fibromatosis. J Foot Ankle Surg 1998;37:227–34. [29] Keus RB, Rutgers EJ, Ho GH, et al. Limb-sparing therapy of extremity soft tissue sarcomas: treatment outcome and long-term functional results. Eur J Cancer 1994;30A:1459 – 63.
Scientific paper
Incidence and treatment of recurrent plantar fibromatosis by surgery and postoperative radiotherapy Eelco de Bree, M.D.a, Frans A. N. Zoetmulder, M.D., Ph.D.a,*, Ronald B. Keus, M.D.b, Hans L. Peterse, M.D.c, Frits van Coevorden, M.D, Ph.D.a a
Department of Surgical Oncology, The Netherlands Cancer Institute/Antoni van Leeuwenhoek Huis, Plesmanlaan 121, 1066 CX Amsterdam, Netherlands b Department of Radiotherapy, The Netherlands Cancer Institute/Antoni van Leeuwenhoek Huis, Amsterdam, Netherlands c Department of Pathology, The Netherlands Cancer Institute/Antoni van Leeuwenhoek Huis, Amsterdam, Netherlands Manuscript received April 29, 2002; revised manuscript November 16, 2002
Abstract Background: The incidence of plantar fibromatosis (PF) is unknown. Sometimes PF tends to recur repetitively after surgical treatment. In our institute we have used postoperative radiotherapy in an attempt to diminish the change on recurrence. Methods: The Dutch Network and National Database for Pathology (PALGA) was consulted to establish the incidence of plantar fibromatosis (PF). Data from 9 patients (11 feet) with PF referred to our institute for recurrent disease were analyzed and the role of postoperative radiotherapy in prevention of recurrence was studied. Results: An average of 1.2 operations for PF was performed per 100,000 citizens yearly in the Netherlands. Twenty-six operations were performed and postoperative radiotherapy was used in 6 cases. Plantar fasciectomy was associated with the lowest recurrence rate. After microscopically incomplete excision or excision of early recurrence (ⱕ6 months) alone all tumors recurred, while recurrence was rarely observed after adjuvant radiotherapy. However, radiotherapy was associated with significantly impaired functional outcome in 3 cases. Conclusions: Plantar fibromatosis is relatively rare. Plantar fasciectomy seems to be the operation of choice. Although effective in decreasing the recurrence rate, adjuvant radiotherapy should be used very selectively because of its serious side effects. © 2004 Excerpta Medica, Inc. All rights reserved. Keywords: Plantar fibromatosis; Incidence; Treatment; Surgery; Radiotherapy
Plantar fibromatosis (PF) is a proliferation of fibroblasts involving the plantar aponeurosis. Fibromatosis is usually localized at sites sensitive to stress, including hand palm, foot, arm, leg, neck, shoulder girdle and abdominal wall. Therefore, it remains unclear whether PF has to be seen as an overreaction to trauma or as a neoplasm. It tends to have a benign clinical behavior with an insidious onset and slowly progressive course; eventually it is locally aggressive with invasion of the overlying skin or deep structures, but metastases do not occur. Considering its clinical behavior, PF has to be seen as a separate entity from aggressive fibromatosis of the extremities [1]. Plantar fibromatosis has usually a milder biological
* Corresponding author. Tel.: ⫹0031-20-5122550; fax: ⫹0031-205122554. E-mail address: [email protected]
behavior. While aggressive fibromatosis tends to recur as skip lesions, each recurrence more proximally, eventual recurrence of PF is usually observed more locally. Generally, PF responds initially to nonoperative measures such as orthoses or corticosteroid injection. Surgery is indicated only if the lesions are highly symptomatic, because surgical treatment is associated with high recurrence rates and significant morbidity [1,2]. We report on the experience with the management of PF at our institute during the last decades. As our hospital is a referral hospital, most cases presented at our hospital with recurrent disease. Analogous to the management of fibromatosis of trunk and extremities [1– 6], adjuvant radiotherapy was given in a number of our cases. The additional use of radiotherapy in PF is mentioned only sporadically in the literature [1,2]. The therapeutic benefit and complications of this approach are discussed. Additionally an effort was made to establish the incidence of operations for PF.
0002-9610/04/$ – see front matter © 2004 Excerpta Medica, Inc. All rights reserved. doi:10.1016/j.amjsurg.2002.11.002
34
E. de Bree et al. / The American Journal of Surgery 187 (2004) 33–38
Patients and methods The Dutch Network and National Database for Pathology (PALGA), including the histological reports from all Dutch pathology departments, supplied information on the incidence of PF in the Netherlands during the period from January 1, 1995, until December 31, 1999. Only total numbers of histological diagnosis of PF could be identified, without differentiating between primary and recurrent disease. A search in the records of our institute over a 20-year period identified 9 patients with PF in 11 feet. The clinical diagnosis was confirmed by histological examination in all but 1 case. In 1 case clinical, radiological, and cytological findings revealed the diagnosis of PF. Not only treatment data from our hospital were gathered, but also data from previous treatment in other hospitals were analyzed retrospectively. Factors identified in the records included age at diagnosis, sex, side of location, eventual period of conservative treatment in our hospital, type and time of primary and secondary surgical treatment, use of adjuvant radiotherapy, treatment complications, and follow-up for recurrence and subjective foot function, starting from time of diagnosis. Additionally, risk factors for development of PF (see discussion) were noted. All operation and histological reports concerning treatment in our hospital as well as in other hospitals were gathered and analyzed. Our pathologists in all cases revised histology from other hospitals. The surgical procedures performed include marginal excision, wide local excision (with at least 1 cm of surrounding macroscopic normal tissue) and subtotal fasciectomy. Excision of the overlying skin and the use of full or split skin grafting were noted. Resections were categorized as macroscopically incomplete, microscopically incomplete, and microscopically complete.
Results Incidence of surgery for plantar fibromatosis in the Netherlands In the 5-year period mentioned, surgical specimens with PF were offered for histological examination 173 to 194 times (average 182) per year in the Netherlands (Table 1). In a country with approximately 15 million citizens, this means an average of 1.2 operations for PF per 100,000 citizens yearly. The age and sex distribution is demonstrated in Table 1, and there were no significant differences in between the consecutive 5 years. Characteristics of patients treated The records of 9 patients, 6 female and 3 male, were studied. Eleven feet, 8 left and 3 right, had PF. The age of the patients at diagnosis varied from 8 to 46 years, with a
Table 1 Age and sex distribution of patients with plantar fibromatosis from whom histological material was obtained in the Netherlands from January 1, 1995, until December 31, 1999 Age (years)
Total
Male
Female
0–9 10–19 20–29 30–39 40–49 50–59 60–69 70–79 80⫹ Total
11 (1%) 26 (3%) 99 (11%) 207 (23%) 239 (26%) 224 (25%) 76 (8%) 27 (3%) 2 (0%) 911 (100%)
6 (1%) 15 (3%) 68 (14%) 153 (31%) 126 (26%) 87 (10%) 26 (5%) 10 (2%) 2 (0%) 493 (100%)
5 (1%) 11 (3%) 31 (7%) 54 (13%) 113 (27%) 137 (33%) 50 (12%) 17 (4%) 0 (0%) 418 (100%)
median and mean age of 25 and 26 years, respectively. Eight patients were referred to our hospital for treatment of recurrent disease (5 feet) or for adjuvant radiotherapy after surgery for local recurrence elsewhere (3 feet). Two patients treated for recurrent disease developed PF at the opposite foot and were subsequently treated again. One patient presented during follow-up examination for treated breast cancer in situ with a nodule of 1.5 cm at the foot sole. Clinical findings and ultrasonography suggested PF. Fine-needle aspiration revealed fibroblastic proliferation, leading to the diagnosis PF. Risk factors Two patients developed bilateral PF, and a 33-year-old patient had a history of multiple excision of a recurrent tumor on her other foot sole when she was 8 years of age. According to this history it might have been also plantar fibromatosis, but we were not able to trace histological data. The same patient had bilateral palmar fibromatosis and a mother with unilateral PF. Another patient developed unilateral palmar fibromatosis during the follow-up period and the father of her mother had been diagnosed with bilateral palmar fibromatosis. Two patients developed PF a few years after local penetrating trauma (needle and metal nail). Primary treatment At diagnosis, all but 3 patients had symptomatic disease. After initial increase in size from 1.5 to 2.5 cm in diameter, spontaneous regression occurred during the next 2 years in 1 patient. At present a lesion of only a few millimeters in size has remained. Two other patients with initially asymptomatic PF were left untreated for periods of 9 months and 3.5 years, until substantial symptoms arose. Absence of response to nonsurgical means necessitated surgical treatment. A total of 26 operations were performed for these 11 sites of PF (Table 2). For primary PF marginal excision was performed in 6 patients, wide local excision in 3 cases, and
E. de Bree et al. / The American Journal of Surgery 187 (2004) 33–38 Table 2 Frequency of recurrence after surgery only and surgery with postoperative radiotherapy
Microscopical completeness of excision Microscopically incomplete Microscopically complete Time to any recurrence and outcome of subsequent treatment of recurrence ⱕ6 months ⬎6 months Surgery for primary plantar fibromatosis Marginal excision Wide local excision Plantar fasciectomy Surgery for recurrent plantar fibromatosis Marginal excision Wide local excision Plantar fasciectomy Below-knee amputation
Surgery only
Surgery and radiotherapy
3/3 12/16
1/4 0/2
3/3 3/6
1/5 0/1
6/6 2/3 1/1
— — —
3/3 2/3 0/1 1/2
0/2 1/3 0/1 —
incompleteness of previous excision necessitated a belowknee amputation. In conclusion, plantar fasciectomy for primary and recurrent PF was associated with the lowest recurrence rate (33%) compared with marginal and wide local excision (82% and 56%, respectively). Value of skin excision Overlying skin was excised in 8 surgical procedures; in only 1 case the skin was microscopically involved. Excluding the last case, because the operation was converted to a below-knee amputation a couple of weeks later, tumor recurred in 4 of the 7 cases after excision of the tumor including overlying skin. A skin transplantation had to be performed to close the defect in 5 cases. Split- and fullthickness skin graft were used each in 2 cases, while in 1 report the type of skin transplant was not mentioned. Adjuvant radiotherapy
plantar fasciectomy in 1 case. Although in only 1 case histological examination demonstrated incomplete local excision of the primary lesion, in 9 primarily surgically treated feet tumor recurred, resulting in a recurrence percentage of (90%). First recurrence was observed 1 to 27 months (mean 9, median 10) after primary surgery. In 1 case asymptomatic recurrence is left untreated until time of this investigation. All other recurrences were operated on. Re-recurrence and its treatment Tumor recurred for the second time in 4 of 8 feet treated surgically for recurrent disease (50%). Treatment and outcome are summarized in Table 3. Marginal excision was performed 5 times for recurrent PF, wide local excision in 6 cases, and plantar fasciectomy in 2 patients. In 1 patient recurrence above the ankle made below-knee amputation necessary. Subsequently, this patient presented with a third recurrence at her knee. After local excision, tumor recurred at the same site for the fourth time and was successfully treated by local excision and radiotherapy. In another case a third recurrence appeared. The local recurrence was treated by microscopically incomplete wide local excision of tumor and overlying skin. In this case, impairment of foot function after previous radiotherapy, wound healing problems, and Table 3 Severe side effects of postoperative radiotherapy in 6 cases Dystrophic foot with impaired foot function Femur fracture†
35
3 cases* 1 case
* In 1 case combination with wound healing problems after microscopically incomplete excision of repeated recurrence necessitated below-knee amputation. † Fourteen years after postoperative radiotherapy for recurrence at knee level.
Adjuvant radiotherapy was applied during the treatment of 6 localizations of PF. In 3 cases 40 Gy was applied to the foot in a generous field reaching to above the ankle with an additional boost of 20 Gy to the foot sole. In 2 cases 60 Gy was given only to the foot sole, while in 1 case of recurrence at knee level 60 Gy was administered to the entire leg remaining after below-knee amputation. In 4 cases it was used after microscopically incomplete excision of recurrent PF, and in another 2 cases as adjuvant treatment after surgery for very early recurrence of PF, 1 and 4 months after initial excision. The results of adjuvant radiotherapy, compared with outcome after surgery only are summarized in Table 2, while the side effects of postoperative radiotherapy are noted in Table 3. Follow-up and functional outcome The mean and median follow-up periods, counted after initial diagnosis of each foot with PF, are 17 and 20 years (range 2 to 26). All patients treated surgically are alive and all but 1 free of disease for the last 13 to 24 years (mean 20 and median 19 months). One patient developed asymptomatic recurrence 2 years ago and is left untreated until today. Two treated feet became dystrophic after surgery and adjuvant radiotherapy and these patients are unable to walk without pain, seriously impairing their quality of life. Two patients are able to walk relatively satisfactory with leg prosthesis after below-knee amputation. The 5 remaining patients retained good leg and foot function.
Comments The exact etiology of PF is unknown. It remains unclear whether PF has to be seen as an overreaction to trauma or as a real neoplasm. A hereditary role has been suggested as
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familial occurrence of plantar and palmar fibromatosis has been reported [7]. Other risk factors reported include history of contralateral plantar fibromatosis, palmar fibromatosis (Dupuytren’s disease) and penile fibromatosis (Peyronie’s disease), trauma, infection, neuropathy, epilepsy, gout, tuberculosis, and chronic intoxication (chiefly lead poisoning) [8 –10]. In the reported series bilateral involvement is observed in a widely varying frequency from 10% to 67% of the patients [1,9 –15]. A wide variation in age at onset exists. In reported series the mean age of treated patients varied from 32 to 49 years [9,12–17]. Our population was significantly younger, but PF has been seen to occur also in childhood [11]. In our Dutch Network and National Database for Pathology, patients operated for PF were most frequently in the fourth to sixth decade of life. Only 4% of patients were younger than 20 years of age and a similar percentage of patients older than 70 years of age. Reports on sex predominance have been contradictory [9 –17]. In our histological archive survey the male-female ratio was 1.2:1. Remarkable is that the highest incidence of histological diagnosis of PF was noted a decade later in females than in males (Table 1). The left foot seems to be slightly more often affected [9 –17]. Plantar fibromatosis generally starts as an asymptomatic nodule in the plantar fascia. Symptoms include pain and swelling on the sole of the affected foot. Despite numerous reports on palmar fibromatosis contractures, similar plantar lesions are rarely observed [18]. Diagnosis is mainly established after physical examination. In an active and early stage, the tumor nodules in tends to be less well defined, softer, and not adherent to the overlying skin, while in a late or resting stage, the lesions is more discrete, harder, commonly adhered to the overlying skin, and measures more than 1 cm. The differential diagnosis includes many malignant and benign soft tissue tumors, including fibrosarcoma and other sarcomas, lipoma, giant cell tumor of tendon sheath, neurofibroma, neurilemmoma, granuloma annulare, rheumatoid nodule, ganglion, epidermoid inclusion cyst, sweat gland carcinomas, and intradermal melanoma [19]. Ultrasonography, computed tomography, and magnetic resonance imaging may visualize size, shape, invasiveness, and exact location [20 –22]. Magnetic resonance imaging is at present considered the modality of choice in the assessment of PF. At histological examination, active proliferation features of fibroblastic cells with many large and hyperchromatic nuclei, loose interstitial collagen fibers, and a mild degree of cellular atypia may be seen. In contrast, in its resting or latent state, large amount of thick and mature fibrous tissue with low cellularity and complete absence of cellular pleomorphism are the common histological features [19,23]. Immunohistochemical examination and ultrastructural analysis reveal similarities with Dupuytren’s disease [24]. The natural history of PF remains one of unpredictable, usually slow, and locally invasive growth. When PF is considered as local fibroblastic proliferation in reaction on
chronic trauma to the plantar fascia, including incorporation of a corpus alienum and insufficient pressure distribution during walking, it is understandable that in most of these cases the lesion remains asymptomatic and stable in size for prolonged periods of time and that spontaneous regression and disappearance occurs [11]. The surgeon is usually consulted for persisting and symptomatic disease. Our survey suggests that approximately one operation for primary or recurrent PF is performed per 100,000 citizens yearly. In other words, a Dutch surgeon operates on PF an average of once in 6 to 7 years. When the lesion recurs after excision, its natural behavior is usually less mild and a more aggressive treatment is warranted. Most of the patients in our series and in other reports belong to the latter group. Nonoperative management is the mainstay of treatment because the disease is often asymptomatic or only mildly symptomatic and responsive to measures such as antiinflammatory medication, modification of activities, physical therapy, orthoses, or occasionally corticosteroid injection [8,10 –15,17,18,23,25,26]. Surgery is indicated only if the lesions are highly symptomatic, locally aggressive, attempts at nonoperative management fail, or if the diagnosis is in question. Surgical treatment is associated with high recurrence rates and significant morbidity [8,10 –14,17,18,23,25]. Although leaving residual disease behind is one of the risk factors for fibromatosis recurrence, microscopically incomplete excision is not automatically followed by local recurrence [2,26,27]. However, in our small series all microscopically incomplete excised tumors recurred, unless adjuvant radiotherapy was administered. Other factors identified with an increased risk for recurrence were multiple nodules, bilateral lesions, and positive family history [12]. When presenting at a young age (ⱕ18 to 30 years), there seems to be a greater likelihood of recurrence after surgery (P ⫽ 0.007 to 0.056) [1,12]. Others support a more favorable course in children [11]. The surgical procedures performed for PF include marginal excision, wide local excision (with a minimal margin of 1 to 2 cm surrounding normal tissue), and subtotal plantar fasciectomy with or without skin grafting, full or split thickness). Different operation techniques have been described for plantar fasciectomy, especially regarding design of the skin incision [8,9,13,15,25]. After local excision of the primary tumor recurrence rates varying from 40% to 100% are reported in different series with probably a negative selection of patients, as discussed above [9,10,12,17]. The high recurrence rate after marginal excision can be explained by the diffuse nature of the lesion along the plantar fascia. In these series, wide local excision and subtotal fasciectomy were more successful as primary treatment, with recurrence rates of 20% to 33% and 0% to 50% respectively [12–17]. Marginal excision as treatment of recurrent disease was associated with a recurrence rate of 70% to 100%, comparable with our results [9,10,12,14]. While others observed an equal failure rate of 75% to 78% after wide local excision of recurrent disease [12,14], Wap-
E. de Bree et al. / The American Journal of Surgery 187 (2004) 33–38
ner et al [13] reported no recurrence after 7 such excisions. It has to be noted that they performed such a wide local excision that this procedure might be called also subtotal plantar fasciectomy in some cases. As also observed in our series, plantar fasciectomy seems to be the most successful procedure and is associated with a failure rate of 0% to 38% [9,12,14]. When PF is seen as local reactive fibrotic tissue due to chronic trauma to the plantar fascia and not as tumor, complete excision of the plantar fascia seems to be logically also the most optimal procedure. Local excision may lead to new fibrotic scar tissue and permits new chronic trauma to the fascia, resulting in high risk on recurrent disease. Complications have been observed up to 18% and 31% of cases after surgery for primary and recurrent PF respectively and include mainly wound healing problems, chronic pain and poor functional outcome [12,13,15]. In only a few studies functional outcome was noted and the results were judged functionally satisfactory in 70% to 80% of cases [12,13,15] In our series functional outcome was good in 60% of treated feet. In the literature, below-knee amputation had to be performed in a widely varying percentage of cases (0% to 22%) for misdiagnosis of fibrosarcoma, more proximal recurrences of aggressive fibromatosis, wound healing problems, severe impaired function of the foot and intractable pain [1,12,13,15]. In 1 of our cases wound healing problems and functional impairment after surgery and radiotherapy necessitated amputation. In another case recurrence to above the ankle made amputation inevitable, but not preventing another recurrence at the knee level. It may be discussed whether this case was more likely to be typical aggressive fibromatosis from the onset rather than PF. Excision of uninvolved overlying skin does not decrease recurrence rate significantly. Involved overlying skin however should be adequately excised and may result in a skin defect, necessitating skin grafting. There is no difference in results or complications between full- and split-thickness skin grafting. Mean time to weightbearing on the affected feet after skin grafting is only slightly longer [12]. Recently, autogenous free deepithelialized dermal fat grafting after primary excision has been proposed as a means to fill up the defect preserving a soft, supple, weightbearing surface with minimal scar tissue formation [28]. Interposition of Marlex surgical mesh after excision of plantar fascia has been advocated for functional restore, but only a small subjective improvement was observed [16]. The use of radiotherapy has been only sporadically reported in the management of PF and therefore its role remains unclear [1,2,9]. Spear et al [1] reported local control of the disease in 2 patients treated by combination of surgery and radiotherapy. Primary radiotherapy as only treatment for recurrent tumor has not been curative in 5 cases reported [1,2,9]. In the treatment of primary and recurrent fibromatosis at other sites, postoperative radiotherapy may result in an increased local control rate [1–5]. While wide local excision with negative margins is the treatment of choice for aggressive fibromatosis, function-sparing resec-
37
tion is appropriate because adjuvant radiotherapy can offset the adverse impact of positive margins [1,4,5]. In our small series, adjuvant radiotherapy was associated with decreased risk for recurrence. After marginal excision, lower extremities remained only free of disease when adjuvant radiotherapy was added. Also after wide local excision radiotherapy resulted in a 50% reduction of the recurrence rate. While all tumors recurred after microscopically incomplete excision only, only 1 of 4 tumors with positive surgical margins recurred when radiotherapy was added postoperatively. When time to recurrence is 6 months or less, subsequent surgery only resulted in recurrence in all cases, while in only 1 of 5 cases recurrence was observed after adjuvant radiotherapy. Radiotherapy doses of 50 to 65 Gy seem to be effective as adjuvant treatment [1– 6,27]. Although adjuvant radiotherapy may result in improved local control, radiation induced impaired function of the foot, lymph edema, marked fibrosis and fracture of irradiated bone might be the price to be paid for this therapeutic benefit [5,29]. Impaired foot function and wound healing after radiotherapy may even lead to amputative surgery. In our series, radiotherapy contributed significantly to the need for amputation in 1 case and resulted in invalidating foot dystrophy in 2 cases. Additionally, the risk for radiation-induced malignancy exists after radiotherapy for this benign disease. In conclusion, marginal local excision regularly leads to local relapse. Plantar fasciectomy is the treatment of choice for primary or recurrent symptomatic PF. Excision of overlying uninvolved skin does not decrease the recurrence rate. Wide excision of extensive disease may result in significant morbidity as a result of excision or damage to adjacent anatomical structures. This implies the need for more local function and structure-preserving procedures in these cases, despite the association with a high recurrence rate. In case of microscopically incomplete surgery or surgery for very early recurrence of PF, adjuvant radiotherapy may diminish the risk on recurrence. However, postoperative radiotherapy of the foot is associated with a high rate of severe adverse effects. In view of its potentially serious side effects, postoperative radiotherapy should be used highly selectively for this benign disease. The risk of complications needs to be weighed against the risk of observation alone. References [1] Spear MA, Jennings LC, Mankin HJ, et al. Individualizing management of aggressive fibromatosis. Int J Radiat Biol Phys 1998;40:637– 45. [2] Miralbell R, Suit H, Mankin HJ, et al. Fibromatoses: from postsurgical surveillance to combined surgery and radiation therapy. Int J Radiat Oncol Biol Phys 1990;18:535– 40. [3] Keus R, Bartelink H. The role of radiotherapy in the treatment of desmoid tumors. Radiother Oncol 1986;7:1–5. [4] Ballo MT, Zagars GK, Pollack A, et al. Desmoid tumor: prognostic factors and outcome after surgery, radiation therapy, or combined surgery and radiation therapy. J Clin Oncol 1999;17:158 – 67.
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