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Foot and Ankle Surgery journal homepage: www.elsevier.com/locate/fas

Review

Osteoid osteoma of the foot and ankle—A systematic review Robert W. Jordan MBChB, MRCSa,*, Togay Koc¸ MBBS MSc MRCSb, Anna W.P. Chapman MBBS MSc(Ed) FRCS (Orth)a, Heath P. Taylor MBBS BSc FRCS (Orth)c a

Birmingham Heartlands Hospital, United Kingdom Southampton General Hospital, United Kingdom c Royal Bournemouth Hospital, United Kingdom b

A R T I C L E I N F O

A B S T R A C T

Article history: Received 23 September 2014 Received in revised form 19 January 2015 Accepted 19 April 2015

Background: Osteoid osteomas are responsible for 10% of benign bone tumours. Treatment typically involves surgical excision or radio frequency ablation. The aim of this systematic review is to evaluate reported cases of foot and ankle osteoid osteomas. Methods: We conducted a systematic review of the literature using the online databases Medline and EMBASE. We included studies reporting osteoid osteoma diagnosed either radiologically or histologically. Results: 94 studies were included reporting 223 cases; 70.5% were male, mean age was 23 years, 69% reported night pain and 72% responded to NSAIDs. The commonest affected bone was the talus. CT scan was the most useful radiological investigation and MRI missed the diagnosis in 34% of cases. The majority of patients underwent surgical excision but an increasing trend of ablation therapy was demonstrated. Conclusions: A high index of suspicion based on salient history and appropriate imaging are essential for timely identification and treatment. ß 2015 European Foot and Ankle Society. Published by Elsevier Ltd. All rights reserved.

Keywords: Foot Ankle Osteoid osteoma Radiofrequency ablation Laser thermocoagulation

Contents 1. 2. 3. 4. 5. 6.

Introduction . . . . . . . . . Material and methods . Results . . . . . . . . . . . . . Clinical presentation . . Discussion . . . . . . . . . . Conclusion . . . . . . . . . . References . . . . . . . . . .

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1. Introduction The osteoid osteoma was first reported by Jaffe in 1935 as a benign osteoblastic bone tumour typically measuring less than 1 cm [1]. Osteoid osteomas are responsible for 10% of benign bone tumours [2] with half of cases involving either the tibia or femur [3]. The talus is the fourth most commonly affected bone occurring

* Corresponding author at: Birmingham Heartlands Hospital, Bordesley Green Eat, Birmingham, B9 5SS, United Kingdom. Tel.: +44 0121 424 2000; fax: +44 0121 424 2200. E-mail address: [email protected] (R.W. Jordan).

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000 000 000 000 000 000 000

in 2–10% of cases [4,5] The other bones of the foot are less frequently involved but include the calcaneus (2.7%), phalanges (2%) and metatarsals (1.7%) [5]. Osteoid osteomas are classified into cortical, cancellous and subperiosteal subtypes [6]. Long bone osteomas tend to be intracortical and are associated with a high amount of subperiosteal reaction. However the majority of foot osteomas are cancellous or subperiosteal where the periosteal reaction is minimal or absent [7]. The pathogenesis is unknown, some postulate it is due to neoplasia [1] and others propose it is an attempt at repair after no apparent injury [8]. A high level of prostaglandins is produced at the centre of the nidus [9] resulting in increased tension and oedema that stimulate nerve endings

http://dx.doi.org/10.1016/j.fas.2015.04.005 1268-7731/ß 2015 European Foot and Ankle Society. Published by Elsevier Ltd. All rights reserved.

Please cite this article in press as: Jordan RW, et al. Osteoid osteoma of the foot and ankle—A systematic review. Foot Ankle Surg (2015), http://dx.doi.org/10.1016/j.fas.2015.04.005

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causing pain. This explains the high level of relief obtained when using non-steroidal anti-inflammatories (NSAIDs). The clinical presentation can vary dependent on the location of the lesion, but a patient with osteoid osteoma can present with tenderness, swelling or effusion, stiffness and/or restricted activities. The classic presentation of night pain and relief with NSAIDs are common findings with one study reporting the latter in 64% of cases [10]. Males are more commonly affected with reported rates of 67–80% as are those under the age of 25 [11,12]. The time taken to obtain the correct diagnosis can be prolonged and has been reported to vary between a few months to years [13]. The rarity of osteoid osteoma and variability of signs on plain radiographs make diagnosis difficult, and therefore, a high index of clinical suspicion is required to help direct further imaging. Foot lesions are commonly located in cancellous bone or peri-articular where periosteal reaction is minimal or absent making radiograph interpretation difficult [14]. Juxta-articular positioning clouds diagnosis further as symptoms replicate those of a monoarthropathy. For example in the talus the condition can mimic ankle impingement, ankle sprain, ostrigonum and inflammatory arthropathy. The typical appearance of an osteoid osteoma on plain radiograph is of a core or nidus cavity of 1–2 cm surrounded by a distinctive zone of reactive bone. However cancellous and subperiosteal lesions often show no periosteal response, although a small lytic erosion may be visualised near the chondro-osseous junction [15,16]. Juxta-articular positioning in the foot makes interpretation difficult due to the complexity of the anatomy. Bone scans have been reported to be positive in 100% of cases [17] but they seldom clearly demonstrate the lesion and often show only generalised uptake. Magnetic resonance imaging (MRI) has been reported to miss the diagnosis in 33–35% of cases [18,19] although oedema within the marrow of the affected bone is usually seen [7]. Computerised tomography (CT) is superior to MRI [7,19] and typically demonstrates a low attenuation nidus with focal central calcification and perinidal sclerosis [20]. The condition can spontaneously regress with reports suggesting the majority resolve between two and six years [21]. However, if symptomatic than surgical excision was the traditional treatment [22,23] with success reported between 88 and 100% of cases [24,25]. En bloc resection with excision of the entire lesion and surrounding reactive zone reduces risk of recurrence but this is associated with a higher degree of bone loss. The use of burr and curettage to remove the surrounding reactive bone is advantageous in limiting the amount of bone loss [26]. Difficulty identifying the nidus intra-operatively may result in either failure to fully excise the lesion or excessive bone excision leading to risk of fracture and necessitating bone grafting. This risk can be reduced with the use of imaging modalities either pre- or intra-operatively to guide excision. Alternative treatments of osteoid osteomas include percutaneous trephine or drilling, ethanol injection and thermal destruction with either laser or radiofrequency ablation. Laser and radiofrequency ablation treatments are performed under radiological guidance with either MR or CT scans. Typically, the procedure is carried out by a team that includes a musculoskeletal radiologist, an anaesthetist and a surgeon. A general or spinal anaesthestic is administered and, under image guidance, entry into the nidus is obtained with either a needle or small drill. The appropriate instrument for ablation is inserted and the position checked under image guidance. Similar results to open procedures have been published with both radiofrequency [27–29] and laser ablation [30,31]. These modalities are less invasive and have the potential advantages of fewer complications and a faster recovery. However, due to the small size of bones in the foot as well as proximity to

neurovascular structures, the use of these modalities has been limited. This systematic review aims to evaluate cases of osteoid osteoma affecting the foot and ankle reported in the literature; analysing patient demographics, symptoms and delay to presentation, diagnostic modalities and treatments for the different bones involved. 2. Material and methods We conducted a systematic review of the literature using the online databases Medline and EMBASE. The search terms used for the Medline search are shown in Table 1 and this strategy was modified when searching EMBASE. The searches were carried out on the 21st May 2014 and were not limited by year of publication. Only papers available in English and with available abstracts were considered for review. We included studies reporting participants who were diagnosed with an osteoid osteoma of any bone of the foot and ankle either radiologically or histologically. Both case reports and case series were included. The study must have reported the location of the osteoid osteoma although variable information on patient demographics and treatment did not preclude study inclusion. Studies were excluded if the location of lesions could not be extracted. In addition, only primary research was considered for review with any abstracts, comments, review articles and technique articles excluded. Eligibility of studies was assessed independently by three authors and any disagreements resolved by discussion. 3. Results Our search strategy revealed 292 studies. After exclusion of duplicates and implementation of inclusion and exclusion criteria 94 studies were included for full paper review [4,7,13,28,30, 32–120]. A flow diagram of this process is demonstrated in Fig. 1. The reasons for exclusion of 100 studies are reported in Table 2. Of the 94 studies included; 64 were case reports [28,32–93] and 30 were case series [4,7,13,30,94–120]. The majority of articles were published after the year 2000 and this chronological frequency is illustrated in Fig. 2. In the 94 studies included for review, 223 cases were reported. The most common bone affected was the talus in 136 cases (61%) as illustrated in Fig. 3. 4. Clinical presentation 70.5% of patients were male and the mean age was 23 years (range 6–64) with the age distribution shown in Fig. 4. Of studies where the age of patients was identifiable, 95% of patient were under the age of 40, and 80% were under 30 years. The majority of patients presented with night pain (69%) although this information was only available in 64% of studies. 72% of patients had found Table 1 Search strategy for Medline. Number

Search term

Results

1 2 3 4 5 6 7 8 9 10

Osteoid osteoma.mp. or exp Osteoma, Osteoid/ Exp Foot/or exp Foot Joints/or Foot.mp. Exp Ankle Joint/or exp Ankle/or Ankle.mp. Calcaneus.mp. or exp Calcaneus/ Talus.mp. or exp Talus/ Exp Metatarsal Bones/or exp Tarsal Bones/ Exp Toe Phalanges/or Phalanges.mp. 2 or 3 or 4 or 5 or 6 or 7 1 and 8 Limit 9 to (abstracts and English language and humans)

2615 109,068 40,519 6983 4486 12,049 3134 135,123 267 126

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MEDLINE

EMBASE

N = 126

N = 166

Combined search

3

98 Duplicates

N = 292

Title review

54 excluded

N = 194

Abstract review

35 Excluded

N = 140

Full paper review

11 Excluded

N = 105 Fig. 1. Flow diagram of review process.

non-steroidal anti-inflammatories to be symptom controlling but this data was only available in 41% of studies. The mean duration of symptoms before diagnosis was 22.1 months (range 1–120 months) with the longest period in talar lesions (see Table 3). There were a variety of radiological investigations used to diagnose the osteoid osteoma. CT was the most commonly used and the most sensitive (96.4%). The results from the other commonly used modalities are shown in Table 4. Of the 94 studies included only one reported using single-photon emission computed tomography (SPECT) [49]. The authors concluded that compared to bone scan alone the technique improved anatomic localisation and gave more precise morphological information. However as the technique was reported in only one study its accuracy could not be compared against other radiological modalities in this review. The commonest treatments overall were en bloc excision and radiologically guided radiofrequency ablation. The proportion of patients receiving the different treatments in each group is shown in Table 5. The mean follow up was 36 months (range 2–131 months) and during this time two

patients had a proven recurrence warranting further treatment (2.4%). Both patients had talar lesions; one patient after an en bloc excision and the other after a CT guided laser thermocoagulation procedure. There was a further complication in that one patient undergoing en bloc excision for a metatarsal lesion suffered a fracture post-operatively.

5. Discussion The talus has previously been reported to be the foot bone most commonly affected by osteoid osteoma [4,5] and the results from this review support this. Jackson et al. also reported involvement of

Table 2 Reason for exclusion of studies after full paper review. Reason for exclusion

Number

Non-foot osteoma Review article Incorrect pathology Not available Incorrect study population Unknown affected bone Conference abstracts Not available in English

34 26 21 7 4 4 3 1

Fig. 2. Included studies according to year of publication.

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R.W. Jordan et al. / Foot and Ankle Surgery xxx (2015) xxx–xxx Table 3 Mean duration of symptoms prior to diagnosis. Bone

Time (months)

Talus Calcaneus Phalanges Metatarsal Cuneiform Cuboid

24 15.8 21 15 18 9

Table 4 Sensitivities (%) of different imaging modalities.

Fig. 3. Pie-chart illustrating the frequency of osteoid osteomas in the foot.

the calcaneus, phalanges and metatarsals [5] which were the next most commonly involved bones in this review. Difficulty and delay in diagnosis has been reported [13]. The present review revealed a mean delay of 22 months between initial presentation and diagnosis, the range was 1–120 months. Although the longest time until diagnosis was seen in the talar group, which may be explained by the complex anatomy of the area, the lower frequency of other bone lesions reduces the information that can be taken from this data. The mean age of all patients was 23 years with 95% under 40 years old. The male to female ratio was just over 2 to 1 which is similar to that previously reported in literature [11,12]. A high proportion of patients reported pain at night (69%) and symptoms relieved by NSAIDs (72%). The presence of these factors in the history should raise the suspicion of an osteoid osteoma to the clinician and appropriate imaging should be obtained. The use of imaging modalities varied greatly between the studies with radiographs, CT, bone scan and MRI all utilised. CT showed the best sensitivity and has previously been reported as superior to MRI in the investigation of osteoid osteomas [7,19]. Although the sensitivity of bone scintigraphy was comparable to CT, it was less specific for osteoid osteoma (typically demonstrating increased uptake) limiting its use to being an adjunct. The failure of MRI to diagnose osteoid osteoma in 34.7% of cases is similar to that reported in the literature [7,18]. The bone marrow oedema signal commonly seen with osteoid osteoma that can be intensely visualised by MRI may mask the typical bony features of the lesion which are nearly pathognomonic on CT. Therefore, MRI appears to lack the specificity to diagnose osteoid osteoma in a significant proportion of patients. This is a concern as MRI is commonly used for the investigation of foot and ankle pain which

All patients Talus Calcaneus Phalanges Metatarsal Cuneiform Cuboid

Plain Radiograph

CT

Bone Scan

MRI

66.4 61.5 60 93.3 80 75 0

96.4 94 94.1 85.7 100 100 100

96.5 94.7 100 100 100 100 –

65.3 74.1 44.4 33.3 37.5 66.7 0

is more commonly attributable to the diagnoses of impingement, osteochondral defects and ligmanentous. We propose that if the patient describes typical history consisting of night pain, response to NSAIDs and is in the at risk age group then the clinician should obtain additional imaging in the form of a CT. A recent study comparing SPECT with CT and bone scans in the diagnosis of osteoid osteomas at all body sites reported that SPECT had a higher sensitivity and specificity (100% and 100%) compared to CT scan (77.8% and 92.3%) and bone scans (100% and 38.4%) respectively [121]. However as only one study [49] in this review reported the use of SPECT, conclusions regarding its accuracy cannot be drawn and further research is still required on this imaging technique, although it may offer a useful adjunct for diagnosis of osteoid osteoma in the foot and ankle. Surgical excision is the traditional treatment [22,23] for osteoid osteomas and is successful in 88–100% of cases [24,25]. More recent developments have led to the availability of thermal destruction with either laser photocoagulation or radiofrequency ablation [27–31]. Although surgical excision remains the most popular treatment modality, both laser and radiofrequency ablation are commonly used despite initial concerns regarding the small size of foot bones and the proximity to neurovascular structures. An increasing trend to using laser and radiofrequency ablation in the treatment of foot osteoid osteomas has been demonstrated with 70.5% of reported cases reviewed in the last five years undergoing one of these two treatments.

Fig. 4. Age of patients included in the review.

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5

Table 5 Different treatment options (%) used for various osteoid osteomas.

Arthroscopic excision Laser thermocoagulation Radiofrequency ablation En bloc excision Excisional biopsy Open curettage Ethanol application

All

Talus

Calcaneus

Phalanges

Metatarsal

Cuneiform

Cuboid

6.3 10.6 25.6 36.9 12.5 7.5 1

10.9 7.6 21.7 38 14.1 6.5 1.1

– 10.7 42.9 32.1 14.3 – –

– 12.5 – 75 12.5 – –

– 7.1 28.6 35.7 – 28.6 –

– 18.2 36.4 45.5 – – –

– 40 20 40 – – –

This systematic review has some limitations; all included studies were either case series or reports and these contribute only level 4 and 5 evidence. The high number of case reports suggests that only rare cases or those undergoing novel treatment may have been publicised which may not give a fair reflection of foot and ankle osteoid osteomas. The data available was restricted to that provided in the manuscript by each author and this varied greatly meaning that each dataset had a variable number of patients. Patients with osteoid osteomas were included for review whether diagnosed histologically or radiologically, therefore some patients may have had the incorrect initial diagnosis. The follow up period was short (mean 36 months); longer and more consistent follow up between the manuscripts would have allowed a fairer reflection on the recurrence rates between the various treatments. However this systematic review analysed a large number of reports and gives an overview of which bones are most commonly involved, which radiological investigations are best and the trends in current management. 6. Conclusion Delay to diagnosis is prevalent in osteoid osteomas of the foot and ankle. This reflects both the high number of differential diagnoses and the difficulties with radiograph interpretation. A high index of suspicion is required in the presence of typical symptoms and an understanding of the appropriate imaging is required for timely identification and treatment. Although surgical excision is the traditional treatment, an increasing trend to treat cases with either laser or radiofrequency ablation is reported. Conflict of interest The authors can confirm that they have no conflict of interests to declare. In addition, no funding was received for the production of this manuscript. References [1] Jaffe HL. Osteoid osteoma. A benign osteoblastic tumor composed of osteoid and atypical bone. Arch Surg 1953;31:709–28. [2] Dahlin DC, Unni KK. Benign osteoblastoma, chapter 8. In: Bone tumors: general aspects of 8,542 cases. Springfield, IL: Charles C Thomas; 1986. [3] Resnick D, Niwayama G. Osteoma. 2nd ed. Diagnosis of bone and joint disorders, vol. 6, 2nd ed. Philadelphia: Saunders; 1988. p. 4081–94. [4] Panni AS, Maiotti M, Burke J. Osteoid osteoma of the neck of the talus. Am J Sports Med 1989;17:584–8. [5] Jackson RP, Reckling FW, Mantz FA. Osteoid osteoma and osteoblastoma. Clin Orthop 1977;128:303. [6] Edeiken J, De Palma AF, Hodes PJ. Osteoid osteoma: roentgenographic emphasis. Clin Orthop 1966;49:201–6. [7] Shukla S, Clarke AW, Saifuddin A. Imaging features of foot osteoid osteoma. Skelet Radiol 2010;39:683–9. [8] Aegerter EE, Kitzpatrick JA. Orthopedic diseases: physiology, pathology, radiology. Philadelphia: PAL Saunders; 1975. 8CC. [9] Makley JT, Dunn MJ. Prostaglandin synthesis by osteoid osteoma. Lancet 1982;2(8288):42. 11CC. [10] Ilyas I, Younge DA. Medical management of osteoid osteoma. Can J Surg 2002;45:435–7.

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Please cite this article in press as: Jordan RW, et al. Osteoid osteoma of the foot and ankle—A systematic review. Foot Ankle Surg (2015), http://dx.doi.org/10.1016/j.fas.2015.04.005