The Foot 31 (2017) 31–34
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Partial fasciectomy is a useful treatment option for symptomatic plantar fibromatosis Haji Khairul Abd Kadir ∗ , Coonoor R. Chandrasekar Royal Liverpool and Broadgreen University Hospitals NHS Trust, Prescot Street, Liverpool, L7 8XP, United Kingdom
h i g h l i g h t s • Plantar fibromatosis is a rare, benign fibroproliferative disorder of the plantar fascia but it can be locally aggressive. • Surgical excision is the mainstay of treatment but recurrence rate is known to be high. • We present a series of 18 patients treated with partial plantar fasciectomy with low recurrence rate.
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Article history: Received 23 January 2017 Received in revised form 11 February 2017 Accepted 16 February 2017 Keywords: Plantar fibromatosis Surgery Recurrence Complications
a b s t r a c t Plantar fibromatosis is a rare, benign fibroproliferative disorder of the plantar fascia. It is considered as a low-grade tumour and it can be locally aggressive. It can present as painful swelling with impairment of local function. Upon failure of non-operative management, surgical treatment options include total fasciectomy or partial fasciectomy. Although surgical excision is the mainstay of treatment, recurrence rate can be up to 60%. The aim of this study was to determine the recurrence and complication rates of surgically treated plantar fibromatosis. A retrospective study was conducted involving patients who had a confirmed diagnosis and excision of plantar fibromatosis treated in our institution between 2011 and 2016. Demographic data, follow-up duration, recurrence and complications were reviewed. Eighteen patients underwent 19 operations. Mean age was 41.3 years (20–57). There were 12 male patients. The main presenting features were pain, swelling and impairment of function. They were investigated by ultrasound scan (12 feet, 63%) and/or magnetic resonance imaging (8 feet, 42%). Two patients (11%) had prior surgery at other institutions whilst 3 patients (17%) had multiple nodules at presentation. All patients underwent partial fasciectomy of the plantar fibromatosis. At up to 5 years follow-up, one patient (6%) had a recurrence in our series whilst 3 patients (17%) had scar related problems (2 patients with scar pain and one patient had hypersensitive scar that resolved after 12 months). Symptomatic plantar fibromatosis can be effectively treated with partial plantar fasciectomy. Crown Copyright © 2017 Published by Elsevier Ltd. All rights reserved.
1. Introduction Plantar fibromatosis, also known as Ledderhose disease, is a rare benign fibroproliferative disorder of unknown origin that can cause nodules to form in the plantar fascia of the foot [1]. It was initially reported by Madelung in 1875 and further described by Georg Ledderhose in 1897 [2,3]. The precise incidence of plantar fibromatosis is unknown but it can occur at all ages [4]. This condition has a preponderance in men than in women [5], and both
∗ Correspondence to: C604 One Park West, 3 Kenyons Steps, Liverpool, L1 3BH, United Kingdom. E-mail address:
[email protected] (H.K.A. Kadir). http://dx.doi.org/10.1016/j.foot.2017.02.002 0958-2592/Crown Copyright © 2017 Published by Elsevier Ltd. All rights reserved.
feet can be affected in 25% of cases [6]. There are many conditions associated with plantar fibromatosis that include long-term diabetes, epilepsy and alcohol consumption [1,7,8]. Dupuytren’s disease (palmar fibromatosis), Garrod knuckle pads and Pyronie’s disease (induratio penis plastica) are strongly associated with this condition [9–11]. Ledderhose disease is viewed as a low-grade tumour but it can manifest as locally aggressive lesion. Clinical features may include insidious onset of pain and swelling in the foot leading to severe impairment of function (Fig. 1). In the early stage, it can present as distension and local pressure which consequently followed by the formation of nodules and contractures of the plantar aponeurosis at the later stages [12]. Critical differential diagnoses such as nodular fasciitis and fibrosarcoma must be ruled out [13], therefore
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Fig. 1. Clinical picture demonstrating a nodule on the plantar aspect of the foot as well as showing a dupuytren’s contracture of the hand.
Fig. 4. T2-weighted magnetic resonance image showing plantar fibromatosis.
Non-operative measures such as the use of orthoses, physical therapy or corticosteroid injections can be helpful during the early phase of the disease [14]. Surgery is indicated when the lesion becomes significantly symptomatic or failed conservative treatment. This can be in the form of partial/limited fasciectomy or total fasciectomy with or without skin grafting. However, it is associated with high recurrence and postoperative complication rates [12,15–18]. According to van der Veer et al., the recurrence rate was estimated to be at 60% when surgical option was utilized [15]. The most common complication after surgery is wound related morbidity [4]. The purpose of this study was to determine the recurrence as well as the complication rates of surgically treated plantar fibromatosis at our institution. 2. Material and methods Fig. 2. Ultrasound scan image showing plantar fibromatosis.
Fig. 3. T1-weighted magnetic resonance image showing plantar fibromatosis.
further investigation in the form of ultrasound scan (Fig. 2) or magnetic resonance imaging (MRI) can help reach the correct diagnosis (Figs. 3 and 4).
A retrospective single-centre study involving patients who had a confirmed diagnosis and excision of plantar fibromatosis treated in our institution between September 2011 and September 2016. Patient data were initially traced by searching our histology records for the diagnosis of plantar fibromatosis and consequently their medical records as well as operative notes were then reviewed. All patients had their lesions surgically removed because they did not respond well to conservative management or had highly symptomatic and debilitating lesions. Information taken from their records included age, gender, duration of symptom, clinical features, family history, mode of investigation, prior surgery, follow-up duration, recurrence and complications were assessed. All of the surgical procedures were performed under general anaesthesia with ankle tourniquet. Partial/limited plantar fasciectomy was performed by removing only the diseased area with a surrounding cuff of normal fascia and the wound was primarily closed after careful haemostasis. Postoperatively, rehabilitation was initiated as per our institution’s protocol. Patients were allowed to weight bear using a well-padded hard soled footwear. The operation notes and histology reports were carefully assessed for consistency and also to ensure that no lesions were identified as fibrosarcoma. None of the patients underwent adjuvant radiotherapy as part of their treatment. Recurrence was defined as reformation of plantar fibromatosis at the surgical site independent of the time period between the resection and the recurrence. This lesion was confirmed by magnetic resonance imaging and histology report.
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Table 1 Results of surgical treatment in 18 patients with plantar fibromatosis. Patient
Age/sex
Solitary
Recurrence/institution
Time to recurrence
Complications
1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18
42/M 33/Ma 20/M 36/F 26/M 37/M 57/M 54/F 44/F 40/M 35/F 30/M 46/F 55/F 55/Mb 49/M 37/M 45/M
Multiple Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Yes Multiple Multiple
No Yes/same No No No No No Yes/other No No No No No Yes/other No No No No
N/A 5 years N/A N/A N/A N/A N/A 1 year N/A N/A N/A N/A N/A 1 year N/A N/A N/A N/A
No No No No No No No No No No Scar pain No No No Scar pain Hypersensitive scar No No
a b
Patient with recurrence in the same institution requiring further surgical intervention. Patient with bilateral presentation.
3. Results Results of surgical treatment in 18 patients with plantar fibromatosis are shown in Table 1. During the 5-year period, 18 patients underwent 19 operations. The mean age was 41.3 years (range: 20–57 years). There were 12 males and 6 female patients. Patients gradually developed symptoms from this disease for a mean period of 25 months (range: 6–72 months). The main presenting features were pain (n = 17), swelling (n = 17) and impairment of function (n = 17). Three patients (17%) confirmed had a family history of plantar fibromatosis. Two patients (11%) had prior surgery at other institutions, presenting with symptomatic recurrent plantar fibromatosis. Fourteen patients (78%) had solitary nodule whilst 3 patients (17%) had multiple nodules at presentation. All of the lesions were located at the medial part of the plantar fascia. The nodules had a mean diameter of 3.2 cm (range: 1.5–7 cm). They were investigated by ultrasound scan (n = 12) and/or magnetic resonance imaging (n = 8). All of the operations were performed by the senior author and all patients underwent partial/limited fasciectomy of the plantar fibromatosis. One patient had bilateral involvement with only one side surgically treated. Sixteen feet underwent surgery for primary lesions whilst 3 feet underwent surgery for recurrent lesions. One patient in the primary group underwent secondary surgical procedure for subsequent recurrent lesion. Postoperatively, patients were reviewed in the outpatient clinic at 2 weeks for removal of sutures, 3 months, 6 months and yearly. All patients had symptomatic improvement and none developed wound infection. Eleven patients (61%) were discharged from clinic at less than 6 months and were advised to return if disease recurred. At up to 5 years follow-up, one patient (6%) had a recurrence which was surgically treated again. None of the patients undergoing second surgical treatment had any further recurrence. 3 patients (17%) had scar related problems whereby 2 patients (11%) had scar pain and one patient developed hypersensitive scar (6%). These patients were referred for pain management/desensitization of the scar. Their symptoms resolved at 12 months and were able to return to full-time employment. 4. Discussion Plantar fibromatosis are common in adult men. They can present as solitary painful nodule, or multiple nodules or thickened cord of plantar fascia. It often involves the medial half of the plantar fas-
cia. Pain and discomfort wearing footwear are common symptoms. Feeling of a pea or a marble inside the shoe is a common description of the patient’s symptom. The disease has a tendency to have insidious onset and slow progression with a mean symptomatic period of 25 months (range: 6–72 months). These findings are comparable to previous reports in the literature [4,15,16,19]. This condition should be investigated further to differentiate it from other serious disease especially fibrosarcoma. Plantar fibromatosis typically appears on ultrasound scan as an isoechoic lesion with a few thin hyperechoic septa that has a heterogeneous internal structure where majority of the swelling adheres to the axis of the plantar fascia (Fig. 2) [20]. On MRI scan, the swelling is usually seen as a well-defined nodule bordering with the plantar fascia with low signal intensity on T1-weighted sequences and low-to-intermediate signal intensity on T2-weighted sequences (Figs. 3 and 4) [20]. On the other hand, a more malignant lesion may show, on ultrasound scan, the presence of vascularisation, inhomogeneity, poor bordering, broad contact with the underlying fascia or invasion of bone or neurovascular bundles [21]. On MRI scan, a mass which demonstrates low signal intensity on T1 weighted images with heterogeneous enhancement after administration of contrast may indicate a more sinister swelling necessitating urgent attention [22]. Operative treatment is generally viewed as a last resort in cases that are refractory to conservative measures. Nevertheless, recurrence rate ranges from 60 to 100% has been reported following local excision of primary lesions [15,16,23]. A wide resection, especially total plantar fasciotomy is believed to be associated with less recurrence [7,8,12,16,23]. However, extensive total plantar fasciectomy is reported to have high morbidity and complications. We believe that partial/limited plantar fasciectomy excising the diseased tissue with a cuff of normal fascia is sufficient to treat plantar fibromatosis with low morbidity and recurrence rates. In this study series, all patients underwent partial/limited fasciectomy and were monitored up to 5 years post operatively. A low recurrence rate of 6% for primary lesion and no further recurrence in three patients with recurrent lesions was recorded. Bilateral feet involvement, multiple nodules and positive family history are described to be associated with increased recurrence rate [19]. However, none of this study patients who presented with recurrence had any of the above factors. Another treatment modality that has been recorded in the literature to reduce recurrence is adjuvant radiotherapy [24,25]. None
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of this study patients had adjuvant radiotherapy treatment. Moreover, foot function was reported to be adversely affected post radiotherapy treatment [4,15]. Radiotherapy was not considered as a treatment option due to low symptomatic recurrence rate in this cohort. Other complications seen in this study were painful scar in 2 patients and sensitive scar in 1 patient. Prior to surgery patients were informed about the risk of local recurrence and sensitive scar. It is important to obtain informed consent, so that the patients are fully aware of the potential risks and benefits of partial/limited fasciectomy. The limitations of the current study are (1) a retrospective study involving a small cohort of patients (2) short term follow-up. However, our results of limited plantar fasciectomy for symptomatic plantar fibromatosis has minimal morbidity with resolution of patient’s symptoms. However, in carefully selected patients, partial/limited fasciectomy is a useful treatment option for symptomatic plantar fibromatosis. Surgery should not be deferred due to presumed high risk of recurrence and wound related morbidity. Radical procedures like total plantar fasciectomy or radiotherapy that is indicated in most cases of plantar fibromatosis, are not recommended. 5. Conclusion Plantar fibromatosis is a rare, benign condition that affects the plantar aponeurosis. Partial/limited plantar fasciectomy is a useful treatment option for symptomatic plantar fibromatosis. Brief summary What is already known • Plantar fibromatosis is a rare, benign fibroproliferative disorder of the plantar fascia. • It is considered as a low-grade tumour and it can be locally aggressive. • Although surgical excision is the mainstay of treatment, recurrence rate is known to be high. What this study adds • A low recurrence rate for primary lesion and no further recurrence in patients with recurrent lesions. • A low complication rate identified and it was mainly due to wound related morbidity. • Radiotherapy was not considered as a treatment option due to low symptomatic recurrence rate in our cohort. References [1] Gordon SD. Dupuytren’s contracture: plantar involvement. Br J Plast Surg 1964;17:412–3.
[2] MO W. Die Aetiologie und die operative Behandlung der Dupuytren’schen Fingerverkrümmung. Berl Klin Wochenschr 1875:191. [3] L G. Zur Pathologie der Aponeurose des Fusses und der Hand. Arch Klin Chir 1897;55:694–712. [4] de Bree E, Zoetmulder FA, Keus RB, Peterse HL, van Coevorden F. Incidence and treatment of recurrent plantar fibromatosis by surgery and postoperative radiotherapy. Am J Surg 2004;187:33–8. [5] Donato RR, Morrison WA. Dupuytren’s disease in the feet causing flexion contractures in the toes. J Hand Surg Br 1996;21:364–6. [6] Haedicke GJ, Sturim HS. Plantar fibromatosis: an isolated disease. Plast Reconstr Surg 1989;83:296–300. [7] Lieberman RA, Ketai N. Plantar fibromatosis. Discussion and illustrative case history. J Am Podiatry Assoc 1973;63:304–9. [8] Skoog T. Dupuytren’s contracture: pathogenesis and surgical treatment. Surg Clin North Am 1967;47:433–44. [9] Meszaros L, Gaspardy G. Simultaneous occurrence of Ledderhose syndrome and Dupuytren contracture. Orv Hetil 1980;121:1775–6. [10] Caroli A, Zanasi S, Marcuzzi A, Guerra D, Cristiani G, Ronchetti IP. Epidemiological and structural findings supporting the fibromatous origin of dorsal knuckle pads. J Hand Surg Br 1991;16:258–62. [11] Snyder M. Dupuytren’s contracture and plantar fibromatosis: is there more than a causal relationship? J Am Podiatry Assoc 1980;70:410–5. [12] Sammarco GJ, Mangone PG. Classification and treatment of plantar fibromatosis. Foot Ankle Int 2000;21:563–9. [13] Galois L, Mainard D, Delagoutte JP. Villonodular tumor mimicking a Ledderhose’s disease. Foot Ankle Surg 2003;9:57–9. [14] Meek RM, McLellan S, Reilly J, Crossan JF. The effect of steroids on Dupuytren’s disease: role of programmed cell death. J Hand Surg Br 2002;27:270–3. [15] van der Veer WM, Hamburg SM, de Gast A, Niessen FB. Recurrence of plantar fibromatosis after plantar fasciectomy: single-center long-term results. Plast Reconstr Surg 2008;122:486–91. [16] Durr HR, Krodel A, Trouillier H, Lienemann A, Refior HJ. Fibromatosis of the plantar fascia: diagnosis and indications for surgical treatment. Foot Ankle Int 1999;20:13–7. [17] Miralbell R, Suit HD, Mankin HJ, Zuckerberg LR, Stracher MA, Rosenberg AE. Fibromatoses: from postsurgical surveillance to combined surgery and radiation therapy. Int J Radiat Oncol Biol Phys 1990;18:535–40. [18] Spear MA, Jennings LC, Mankin HJ, Spiro IJ, Springfield DS, Gebhardt MC, et al. Individualizing management of aggressive fibromatoses. Int J Radiat Oncol Biol Phys 1998;40:637–45. [19] 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. [20] Omor Y, Dhaene B, Grijseels S, Alard S. Ledderhose disease: clinical, radiological (ultrasound and MRI), and anatomopathological findings. Case Rep Orthop 2015;2015:741461. [21] Widmann G, Riedl A, Schoepf D, Glodny B, Peer S, Gruber H. State-of-the-art HR-US imaging findings of the most frequent musculoskeletal soft-tissue tumors. Skeletal Radiol 2009;38:637–49. [22] Demir HA, Akyuz C, Varan A, Ergen FB, Buyukpamukcu M. Right foot congenital infantile fibrosarcoma treated only with chemotherapy. Pediatr Blood Cancer 2010;54:618–20. [23] Allen RA, Woolner LB, Ghormley RK. Soft-tissue tumors of the sole; with special reference to plantar fibromatosis. J Bone Joint Surg Am 1955;37-A:14–26. [24] Heyd R, Dorn AP, Herkstroter M, Rodel C, Muller-Schimpfle M, Fraunholz I. Radiation therapy for early stages of morbus Ledderhose. Strahlenther Onkol 2010;186:24–9. [25] Seegenschmiedt MH, Attassi M. Radiation therapy for Morbus Ledderhose — indication and clinical results. Strahlenther Onkol 2003;179:847–53.