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Foot and Ankle Surgery xxx (2017) xxxxxx

Contents lists available at ScienceDirect

Foot and Ankle Surgery


journal homepage: www.elsevier.com/locate/fas

Case report

A rare cause of lateral ankle pain: A symptomatic talus secundarius


Ruben Zwiers, M.D.a,b,c,* , Peter A.J. de Leeuw, M.D.a,b,c ,
Gino M.M.J. Kerkhoffs, M.D. Ph.D.a,b,c , C. Niek van Dijk, M.D. Ph.D.a,b,c
a
Academic Medical Center, Department of Orthopaedic Surgery, Amsterdam, The Netherlands
b
Academic Center for Evidence-Based Sports Medicine (ACES), The Netherlands
c
Amsterdam Collaboration for Health and Safety in Sports (ACHSS), The Netherlands

A R T I C L E I N F O A B S T R A C T

Article history:
Received 23 December 2016 The talus secundarius is one of the rarest accessory tarsal bones, being present in 0.01% of all ankles. It is
Received in revised form 8 March 2017 located at the lateral side of the talus, distally to the tip of the bula. Hitherto, only ve cases of a
Accepted 21 March 2017 symptomatic talus secundarius have been described in literature. We presented a case of bilateral
Available online xxx symptomatic talus secundarius in a young gymnast. There was a difference in size of the two accessory
bones. The large talus secundarius in the left ankle was xated, in the right ankle the fragment was
Keywords: excised. Both excision and xation in the presented patient led to satisfactory results, both in the short
Ankle and long term outcome.
Arthroscopy
2017 Published by Elsevier Ltd on behalf of European Foot and Ankle Society.
Accessory bones

1. Introduction and eversion in the talocalcaneal joint. There was recognizable


tenderness on palpation at the anterolateral aspect of both talar
The os trigonum and the os tibiale externum are the most bones, specically 1 cm distally to the anterolateral ligament
common accessory bones in the foot and ankle. The talus complex. Both the anterolateral ankle ligaments and the syndes-
secundarius is generally less known. It is located at the lateral mosis were stable. In addition, there was tenderness over the distal
side of the talus, distally to the tip of the bula and is one of the aspect of the peroneal tendons, just distally to the tip of the lateral
rarest accessory ankle bones, being present in 0.01% of all ankles malleolus. However, peroneal tendon resistance tests did not
[1]. Up to present only ve cases of a symptomatic talus reveal abnormalities. Based on the patient complaints and the
secundarius have been described in literature [25]. We performed physical examination the differential diagnosis included a stress
a literature review on this rare accessory bone and present a case of fracture, peroneal tendon instability, or a subtalar coalition.
bilateral symptomatic talus secundarius in a high-level athlete. The computed tomography (CT) scan of the right ankle showed
a small rounded fragment (7  5  2 mm) lateral to the talus at the
2. Case report level of the subtalar joint, forming a synchondrosis with the talus.
At the same location in the left ankle a larger osseous fragment was
A fourteen years old high-level gymnast presented with found (17  15  10 mm), with small cysts at the level of the
increasing pain at the lateral aspect of both ankles mainly during synchondrosis (Fig. 1). In both ankles the superomedial aspect of
jumping exercises. The patient could not recall any trauma the calcaneus showed an atypical aspect. In addition magnetic
preceding his current complaints. With normal daily activities resonance imaging (MRI) showed bilateral bone marrow edema of
no limitations were reported. Past medical history did not reveal the talus at the level of the fragments (Fig. 2).
any other foot and ankle pathologies. On physical examination The patient was diagnosed with a bilateral symptomatic talus
there was a normal hindfoot alignment, the range of motion of both secundarius. Initially, he was treated nonoperatively for a period of
the tibiotalar and talocalcaneal joints were symmetric, being 20 of 6 weeks predominantly with rest and immobilization in a Walker
dorsiexion and 40 of plantar exion and 20 of both inversion boot. On follow-up there was no improvement and therefore it was
decided to surgically address the pathology.
The patient underwent the surgical procedure for both ankles in
a single session, under spinal anesthesia in the daycare unit.
* Corresponding author at: Academic Medical Center, University of Amsterdam,
Patient was positioned supine with a tourniquet around both
Department of Orthopedic Surgery, P.O. Box 22660, 1100 DD Amsterdam, The
Netherlands. Fax: +31 205669117. upper legs and prophylactic antibiotics (cefuroxime iv) were given
E-mail address: r.zwiers@amc.uva.nl (R. Zwiers). according to the local protocol. At the right side the bony fragment

http://dx.doi.org/10.1016/j.fas.2017.03.011
1268-7731/ 2017 Published by Elsevier Ltd on behalf of European Foot and Ankle Society.

Please cite this article in press as: R. Zwiers, et al., A rare cause of lateral ankle pain: A symptomatic talus secundarius, Foot Ankle Surg (2017),
http://dx.doi.org/10.1016/j.fas.2017.03.011
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Fig. 1. Computed tomography of both ankles showed a bilateral talus secundarius. A and C: right ankle small rounded ossicle (7  5  2 mm). B and D: large fragment
(17  15  10 mm) with small cysts at the level of synchondrosis.

was removed through a mini-open surgical approach, just distally up the right side was allowed weight bearing as tolerated without
from the tip of the lateral malleolus. The anterior talobular immobilization. The left ankle was immobilized for another eight
ligament and the talocalcaneal ligament remained undisturbed. At weeks, consisting of a four weeks below knee weight bearing cast
the left side it was decided to rigidly xate the talus secundarius and four weeks with a Walker. A prophylactic dose of low
onto the talar body because of its signicant contribution to the molecular weight heparin was prescribed for 6 weeks to prevent
talocalcaneal surface area. A similar surgical approach was thromboembolic events.
performed as in the right ankle and both the surface of the bony At three months follow up, the right ankle had fully healed,
fragment and the talus were debrided, without the need to release whereas the left ankle was still tender on palpation over the
the anterolateral ankle ligaments. Compression and xation were anterolateral talus. The wound on both ankles healed uneventfully
achieved by means of two small fragment lag screws (26 mm and there was no injury to the neurovascular structures. Both
2.5mm). ankles had a full range of motion, and the patient was allowed to
Postoperative management for both ankles consisted of non- resume sporting activities as tolerated under the supervision of the
weight bearing below knee casts for 2 weeks. At two weeks follow sports physician and physiotherapist. Postoperative weight

Please cite this article in press as: R. Zwiers, et al., A rare cause of lateral ankle pain: A symptomatic talus secundarius, Foot Ankle Surg (2017),
http://dx.doi.org/10.1016/j.fas.2017.03.011
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Fig. 2. Magnetic resonance imaging of both ankles. A and C: right ankle, small talus secundarius fragment (red arrow) with bone marrow edema at the level of the fragment. B
and D: left ankle, talus secundarius (red arrow) with bone marrow edema at the level of the bony fragment.

bearing radiographs of the left ankle showed signs of consolidation 3. Discussion


(Fig. 3). At one year follow up there was occasionally some
tenderness in the left ankle during intensive activity, which Talus secundarius is very rarely a symptomatic pathology
however did not prevent him from performing at high level needing (surgical) treatment. In this study we present a case of
gymnastics. At two years follow up he was free of pain during all bilateral symptomatic talus secundariius. Both excision and
activities (numeric rating scale (NRS) for pain 0/10) and his xation in the presented patient led to satisfactory results, both
function was equal to the pre-injured situation (NRS function 10/ in the short and long term outcome.
10; FAOS scores: symptoms 88, pain 100, activity 100, sport 100, The term talus secundarius was rstly used by Gruber in 1864
quality of life 100). At nal follow-up, seven years postoperatively, [6]. He mentioned the accessory bone posterior to the talus,
his condition was unchanged. Patient was satised with the result already described by Rosenmller in 1804 [7], which currently is
of both surgeries (NRS satisfaction 8/10) and declared he would known as the os trigonum. Ptzner was rst to describe an
undergo the same surgery again in similar circumstances. accessory bone lateral to the lateral process of the talus and

Please cite this article in press as: R. Zwiers, et al., A rare cause of lateral ankle pain: A symptomatic talus secundarius, Foot Ankle Surg (2017),
http://dx.doi.org/10.1016/j.fas.2017.03.011
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Fig. 3. Postoperative weight bearing radiographs of the left ankle, AP (A) and lateral (B), three months after surgery, showed adequate position of the screws and early signs of
consolidation.

Table 1
Overview of published cases of symptomatic talus secundarius. F = female, M = male.

Study Age Sex Side Symptoms Phys. exam Etiology Connection talus Treatment
Schluter [4] 12 F Right Ankle sprain Synchondrosis Resection
16 M Left Ankle sprain Synchondrosis Resection
Hahn [3] 25 M Right Lateral pain uneven Tenderness bula tip, No trauma Synostosis Resection
ground normal peroneal tendon
function
Viana et al. [2] 31 M Right Lateral ankle pain aggravated Synostosis
by walking and standing
Oliveira et al. [5] 18 M Bilateral Ankle pain which was Tender point just distal No trauma Synchondrosis Resection
aggravated by sports to both bular malleoli

considered it as the result of an unhealed fracture [8]. After


Bardeleben introduced the term os trigonum for the posterior
fragment [9], the name talus secundarius was available for the
lateral located fragment, as described by Ptzner [8]. In literature
the term talus secundarius is still sometimes erroneously used
when referring to the os trigonum or the talus accessorius. The
talus accessorius is located at the medial side of the talus [8]. The
term talus secundarius is also used for other accessory ossicles as
well, like the os supratalare or the os subtibiale [10,11].
Radiologically the talus secundarius can be confused with the os
subbulare, an accessory bone that is located distally to the bula
[2]. Furthermore, it must be distinguished from a lateral talar
process fracture [12] (Fig. 4).
The talus secundarius is one of the rarest accessory bones of the
foot. In a large Japanese study, three ankles with a talus
secundarius were detected, resulting in a prevalence 0.01% [1].
The rst cases of symptomatic talus secundarius have been
described in 1952 by Schluter [4]. She described two cases of
symptomatic talus in young patients after an ankle sprain. Since
then only three additional cases have been published [2,3,5].
Fig. 4. Accessory bones of around the talus, according to Ptzner [8]. 1 = os talus
Patients aged from 12 to 31 years old. In these published cases ve secundarius, 2 = os subbulare, 3 = calcaneus secundarius, 4 = os supratalare, 5 = os
out of six patients were males, whereas in the radiological study trigonum, 6 = os talus accessorius.

Please cite this article in press as: R. Zwiers, et al., A rare cause of lateral ankle pain: A symptomatic talus secundarius, Foot Ankle Surg (2017),
http://dx.doi.org/10.1016/j.fas.2017.03.011
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one of the three patients was male. In just two out of six size and thereby in its contribution to the talocalcaneal joint
symptomatic cases a clear injury occurred prior to the onset of surface area. Symptomatic patients may suffer from lateral ankle
symptoms. Only one other case of bilateral talus secundarius was pain during activity. Depending on the size of the fragment and
found [5]. The bilateral prevalence is unknown. Four of the involvement of the subtalar joint, either excision or xation of the
published cases underwent excision of the fragment, all with good fragment can be performed. In the presented case both
results (Table 1). interventions have led to satisfactory results both in the short-
The origin of the talus secundarius is unclear. Following the and long term.
current literature the talus ossies from two ossication centers,
the main talar body and the posterior process. There is no
literature on the existence of other ossication centers [13]. It is Conict of interest
however hypothesized that accessory bones are the result of
remnants of elements that arise during the ontogenesis of the All authors state not to have conict of interest or something
talus [14]. There has been several theories on the development of else to disclose.
the tarsal bones, of which a commonly used theory is that the
tarsal bones are formed out of the different canonical elements,
as described by Gegenbaur. These consists of the tibiale,bulare, References
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Please cite this article in press as: R. Zwiers, et al., A rare cause of lateral ankle pain: A symptomatic talus secundarius, Foot Ankle Surg (2017),
http://dx.doi.org/10.1016/j.fas.2017.03.011

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