The Journal of Foot & Ankle Surgery xxx (2017) 16

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The Journal of Foot & Ankle Surgery

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Case Reports and Series

Massive Osteochondral Lesion of the Talus in a Skeletally Immature

Patient Associated With a Tarsal Coalition and Valgus Hindfoot
Pablo A.I. Slullitel, MD, Maria L. Tripodi, MD, Santiago T. Bosio, MD, Miguel Puigdevall, MD,

n Maenza, MD
Rube
Orthopaedist, Institute of Orthopaedics Carlos E. Ottolenghi, Italian Hospital of Buenos Aires, Buenos Aires, Argentina

a r t i c l e i n f o a b s t r a c t

Level of Clinical Evidence: 4 Rarely, osteochondral lesions of the talus occur without a history of trauma. Accurate interpretation of the
mechanical load distributions onto the ankle leading to potential atraumatic cartilage damage must always be
Keywords:
hindfoot malalignment studied. The published data on the optimal treatment of talar osteochondral lesions in skeletally immature
osteochondral allograft transplantation patients are scarce, especially when the lesions are associated with hindfoot malalignment. We describe the
osteochondral lesion of the talus case of a pediatric female with an atraumatic osteochondral lesion of the talus associated with a talocalcaneal
talocalcaneal coalition coalition and a valgus hindfoot, which we consider the rst case to be reported. She presented with prolonged
valgus hindfoot bilateral ankle pain and catching during gait of approximately 2 years duration with a restricted range of
motion, with the pain more excruciating in the right ankle. Radiographs revealed a large osteochondral lesion
located at the lateral talar dome. The patient underwent partial osteochondral allograft transplantation,
together with hindfoot realignment and coalition resection with a fat graft interposition. At the 2-year follow-
up examination, the patient was free of pain in her right foot and ankle, with no signs of radiologic failure.
2017 by the American College of Foot and Ankle Surgeons. All rights reserved.

Similar to those in adults, osteochondral lesions of the talus (OLTs) atraumatic bilateral OLT related to a talocalcaneal coalition and valgus
in skeletally immature patients are usually secondary to traumatic hindfoot, which we consider the rst case to be reported. Because the
episodes to the ankle such as sprains or fractures (1). Chondral in- published data on the optimal treatment of OLTs in skeletally
juries have been reported to be as great as 73% after ankle fractures immature patients are scarce, we believe our case report emphasizes
(2), with the posteromedial and anterolateral aspects of the loading the straight relationship between the ankle biomechanics and
surface of the talus the most commonly affected areas (3). Tradi-
tionally, the diagnosis of OLTs is made after a traumatic event using
plain radiographs, magnetic resonance imaging (MRI), and physical
examination, which could reveal swelling, a decreased range of mo-
tion, catching or locking, and claudication.
Rarely, OLTs occur without a history of acute or chronic trauma (4).
Sometimes, medical issues such as long-term steroid therapy, endo-
crine abnormalities, or embolic disease or congenital factors such as
ligamentous laxity can be the major associated conditions, especially
when the osteochondral lesion is bilateral (47). However, accurate
interpretation of the mechanical load distributions onto the ankle
leading to the potential for talar cartilage damage must also be
studied primarily, given that hindfoot malalignment and a tarsal
anatomic alteration could probably aid in the pathologic entitys
endurance. We describe the case of a pediatric patient with an

Financial Disclosure: None reported.

Conict of Interest: None reported.
Address correspondence to: Pablo A.I. Slullitel, MD, Institute of Orthopaedics Fig. 1. Clinical image of patients feet seen from a posterior view showing a rigid bilateral
Carlos E. Ottolenghi, Italian Hospital of Buenos Aires, Buenos Aires 1199, Argentina. valgus hindfoot, associated with bilateral forefoot abduction, which is more evident on
E-mail address: pablo.slullitel@gmail.com (P.A.I. Slullitel). the right foot.

1067-2516/$ - see front matter 2017 by the American College of Foot and Ankle Surgeons. All rights reserved.
http://dx.doi.org/10.1053/j.jfas.2017.04.025
2 P.A.I. Slullitel et al. / The Journal of Foot & Ankle Surgery xxx (2017) 16

Fig. 2. (A) Anteroposterior and (B) lateral radiographs of the patients right ankle showing a large osteochondral lesion located at the lateral talar dome, classied as stage V of the
modied Berndt and Harty classication (11). (C) Computed tomography axial image of the right ankle showing a well-circumscribed, cystic osteochondral lesion (D) Magnetic resonance
sagittal image of the same ankle depicting the cystic chondral damage and lucencies, with a large area of edema underneath, classied as stage V using the Hepple classication (12).

obtaining successful joint-preserving treatment when a massive OLT
has been diagnosed.
Case Report
During January 2014 and at age 13 years, an otherwise normal
female came to our institution because of prolonged bilateral ankle
pain and catching during gait of approximately 2 years duration. The
symptoms were denoted to be in crescendo, with difculty per-
forming, and sometimes an inability to perform, the activities of daily
living. The patient reported the spontaneous onset of pain and
swelling without a denite traumatic event.
On physical examination, palpation revealed tenderness primarily
on the lateral aspects of both ankles that was more excruciating with
the joint in maximal plantarexion. During passive exion and exten-
sion of the ankle, joint locking was both palpable and audible. In all
tests, the right ankle proved to be more symptomatic. The range of
motion measured using a traditional goniometer showed a slightly
more restricted right ankle (plantarexion 0
to 45
; dorsiexion 0
to
15
) compared with the left ankle (plantarexion 0
to 50
; dorsi-
exion 0
to 20
), without evidence of stiffness. Joint laxity was
 P.A.I. Slullitel et al. / The Journal of Foot & Ankle Surgery xxx (2017) 16 3

Fig. 3. (A) Computed tomography coronal image of the patients right foot with a shingled-type (type 3) talocalcaneal coalition using Mubaraks grading system (13). (B) Computed
tomography coronal image of the patients left foot showing a complete osseous bar (type 4) using Mubaraks classication.

certied by the anterior drawer test ndings, and stability was assessed
using the star excursion balance test (8). No evidence of hypermobility
or instability was found after thorough examination. Furthermore, a
rigid bilateral valgus hindfoot was appreciated (Fig. 1). Its lack of
correction with toe standing (heel rise test [9,10]) was suggestive of
limited subtalar motion. Additionally, it was associated with bilateral
forefoot abduction, which manifested more in the right foot (Fig. 1).
Radiographs revealed a bilateral large OLT located at the lateral
talar dome. Further imaging with computed tomography and MRI
showed a well-circumscribed, deep, cystic OLT with lucencies and
edema underneath (Fig. 2). We classied the OLT as stage V of the
modied Berndt and Harty classication (11) from the ndings on
plain radiographs and stage V of the Hepple classication (12) from
the MRI ndings. Additionally, a bilateral talocalcaneal coalition was
detected on the computed tomography images (Fig. 3). These were
classied as a complete osseous bar (type 4) on the left side and a
shingled one on the right (type 3), using Mubaraks grading system
(13). The hindfoot alignment angle was 15
in the right foot and 18
in
the left (14).
Because the longevity of the symptoms and the incapacitating pain
were primarily present in the right ankle, surgical treatment was
indicated rst for this joint. With the patient under epidural hypo-
tensive anesthesia and placed in the supine position with a supra-
patellar tourniquet, an anterolateral approach with a bular malleolar
osteotomy was performed. The bular osteotomy was executed
proximal to the anterior tibiobular ligament. After gentle manual
distraction, the ankle joint was fully visualized. A 3.6-cm2, well-
circumscribed, cystic OLT was found in the lateral talar dome. The
lesion was resected en bloc through a minimal osteotomy to preserve
as much of the native talus as possible, until bleeding bone was
exposed (evaluated by temporarily deating the tourniquet). The
subchondral cysts were curetted and lled with fresh cancellous
allograft. The size of the lesion was meticulously measured, and a
matching graft (partial structural osteoarticular allograft) of the same
size and shape was obtained from the same anatomic region of a
donor talus (Fig. 4). The graft and bed of the lesion were accurately
contoured such that an anatomic press t could be obtained. Graft
xation was achieved with 2 headless Herbert (Zimmer, Warsaw, IN)
4 P.A.I. Slullitel et al. / The Journal of Foot & Ankle Surgery xxx (2017) 16

Fig. 4. (A) Intraoperative image of the right ankles arthrotomy using an anterolateral approach with a lateral malleolar osteotomy showing a macroscopic view of the lesion, with
numerous cysts located on the lateral aspect of the talar dome, which could be seen on previous complementary imaging studies. (B) Intraoperative image after meticulous resection of
the osteochondral lesion until a bleeding bed of remaining bone was seen with release of the tourniquet. (C) Clinical image of fresh-frozen donor talus after the lesion size had been
measured to match the size and shape to that obtained after excision. (D) Image showing incorporation of the structural graft into the embedded remaining lesion, accurately contoured to
obtain an anatomic press t.

screws using intraoperative uoroscopic guidance. After joint irriga-
tion, the lateral malleolar osteotomy site was stabilized with a 5-hole
one-third semitubular plate with screws, using a lag screw through
one of the holes to compress the osteotomy line (Fig. 5).
Subsequently, resection of the tarsal coalition with fat graft inter-
position was performed, as previously described by Olney and Asher
(15) and modied by Gantsoudes et al (16). Using a minimally invasive
medial approach, a coalition resection was performed with an
osteotome and a high-speed 4-mm burr, with assessment of the
hindfoot motion under uoroscopic guidance. The fat graft used was
taken from the previous anterolateral incision, with the addition of
bone wax over the bony surfaces of the talus and calcaneus. Finally, to
correct the valgus-positioned hindfoot, a calcaneus varus osteotomy
was completed through a percutaneous approach, as previously re-
ported (17). The desired correction was controlled under an image
intensier through the axial and sagittal views and xed with 2
cannulated partially threaded screws.
The pathologic examination of the biopsy specimen revealed focal
areas of articular damage with a diffuse absence of cartilage
sheathing, along with cystic areas of necrosis and chronic inam-
mation of the subchondral bone. Intraoperative cultures were nega-
tive for infection. Postoperatively, the ankle was immobilized for 15
days and then placed in a removable walking boot to allow for early
range-of-motion exercises. Weightbearing was started 8 weeks after
surgery, after graft integration was evidenced on the follow-up ra-
diographs. The patient was examined and radiographs were obtained
at 8 weeks and 6 months postoperatively and then annually. The
calcaneal screws were surgically removed at 6 months post-
operatively because of implant-related symptoms during gait.
At the 2-year follow-up examination, the patient was free of pain
in her right foot and ankle, with no signs of radiologic failure (Fig. 6).
Given that symptoms had dramatically increased in the left foot, the
same surgical treatment was indicated, which the mothers patient
and the patient herself accepted without concern. The recorded
American Orthopaedic Foot and Ankle Society (AOFAS) Ankle-
Hindfoot scale score (18) showed an increase from the preoperative
score of 45 to a postoperative follow-up score of 92. The visual analog
scale for pain score also showed an improvement from 10 points
preoperatively to 0 points during the postoperative follow-up period.
Discussion
Atraumatic OLTs represent a surgical challenge to orthopedic
surgeons, especially in the pediatric population for whom good to
excellent long-term outcomes are even more important. Despite the
rarity of OLTs (4), a thorough physical examination remains a requisite
because, as we have shown, OLTs can be associated with a mechanical
imbalance that could jeopardize, if not addressed, the endurance of an
eventual treatment of the talar cartilage.
In the present case, we were not able to conrm that the tarsal
coalition and valgus hindfoot were the result of the OLT; many un-
derlying factors could have been involved in its development, including
 P.A.I. Slullitel et al. / The Journal of Foot & Ankle Surgery xxx (2017) 16
Fig. 5. Radioscopic image of the patients right foot. Graft xation was achieved with 2
Herbert screws, and the lateral malleolar osteotomy site was stabilized with a 5-hole
one-third semitubular plate with screws using a lag screw through 1 of the holes to
compress the osteotomy line. Additionally, the calcaneal varus osteotomy was per-
formed percutaneously and xed with 2 cannulated partially threaded screws under
uoroscopic guidance.
Fig. 6. (A) Anteroposterior and (B) lateral radiographs of the patients right ankle at 2 years postoperatively, with no signs of secondary osteoarthritis or allograft resorption.
5
endocrine, genetic, vascular, or even congenital abnormalities, espe-
cially considering that the patient had bilateral OLTs (47). We believe
the etiology of this OLT remains unknown. Nonetheless, an overload on
the lateral aspect of the talus provided by the 15
valgus hindfoot over a
rigid subtalar joint might have mechanically predisposed to the origin
or even aggravation of an OLT on its lateral aspect (19,20). Paul et al (21)
retrospectively analyzed the data from 22 patients who had undergone
autologous osteochondral transplantation for an OLT and assessed the
relationship between OLT localization and hindfoot alignment. They
found no association (p .766) between the hindfoot alignment and
the location of the OLT and suggested that other factors (metabolic,
vascular, idiopathic) might play a role. However, they had studied a
small cohort of patients without a control group, and none of the pa-
tients included had a xed (tarsal coalition) subtalar joint, which might
contribute to talar overload (19,22).
The correction of hindfoot malalignment seems to play a major
role in the treatment of talocalcaneal coalitions and other associated
pathologic entities. Mosca and Bevan (22) reported 13 patients with
painful talocalcaneal coalitions who underwent calcaneal length-
ening (and valgus-correcting) osteotomy. They concluded that treat-
ment of valgus deformity appears to be as important as treatment of
the coalition itself, obtaining good to excellent results at mid-term
follow-up. In a case similar to ours, Hotel and Engelhardt (23) re-
ported the case of a young alpine ski racer with osteochondritis dis-
secans of the talus. In addition to an autologous matrix-associated
chondrogenesis as baseline treatment, they performed a lateral
calcaneal distraction osteotomy to correct the hindfoot malalignment.
At the nal follow-up examination, the patient had returned to the
previous level of training (23).
The published data on the optimal treatment of OLT in skeletally
immature patients are scarce. Conservative management (24) has
6 P.A.I. Slullitel et al. / The Journal of Foot & Ankle Surgery xxx (2017) 16
been proposed for stage I and II of the Berndt and Harty classication
(11). In cases of failure of conservative treatment or more advanced
stages, surgical treatment remains the reference standard. Similar to
the knee, bone marrow stimulation through microfractures (open or
arthroscopic) and debridement of unstable cartilage aps are usually
performed. However, the overall success rate of the treatment de-
pends on the stage of the lesion (25). Although stage I and II are
treated with bone marrow-stimulation techniques, osteochondral
autografts or allografts and autologous chondrocyte implantation or
related procedures are used for stage IV or greater. In the present case,
given the magnitude of the OLT, we decided to perform fresh talar
osteochondral allograft transplantation. Despite the technical dif-
culty of fashioning the graft into a specic radius of curvature, we
believe this should be the standard treatment for cases of cystic OLTs
3000 mm3 in size, as previously stated (26).
Raikin (27) prospectively evaluated 15 patients with a mean
follow-up period of 44 months (minimum 24 months) after fresh bulk
osteochondral allograft transplantation for large-volume OLTs. At the
nal follow-up examination, the mean AOFAS Ankle-Hindfoot scale
score had improved from 38 points preoperatively to 83 points
postoperatively, and visual analog scale for pain score had decreased
from a mean of 8.5 points preoperatively to 3.3 points postoperatively.
Radiographic ndings revealed evidence of collapse or resorption in
10 ankles (67%); however, 2 of the 15 ankles (13%) required conversion
to ankle arthrodesis. In a similar study, Haene et al (28) reported a
reoperation rate of 12% and failure rate of 29% after prospectively
evaluating 17 cases of fresh osteochondral talar allograft trans-
plantation. Very few studies have been reported on the outcomes of
allograft transplantation in the medium or long term specically in a
pediatric population (29). Only Raikin (26) reported on 2 pediatric
patients who each had obtained an AOFAS Ankle-Hindfoot scale score
of 100 points at the 2-year follow-up examination.
In conclusion, although speculative, we believe that the endurance
of the fresh bulk osteochondral allograft transplantation might be
guaranteed by both the hindfoot realignment and resection of the
talocalcaneal coalition. First, an aligned hindfoot would avoid any
overload over the implanted allograft and, second, the coalition
excision would aid in maintaining subtalar motion and preventing the
early development of ankle osteoarthritis (30). Considering that most
pediatric patients undergoing bone marrow stimulation procedures
have good outcomes over time (31), those patients who undergo
osteochondral allograft transplantation should be chosen carefully.
We use this treatment for selected patients, because it has the po-
tential to require revision surgery, such as ankle arthrodesis or total
ankle arthroplasty, at a still undetermined follow-up point.
References
1. Giannini S, Buda R, Vannini F, Cavallo M, Grigolo B. One-step bone marrow-derived
cell transplantation in talar osteochondral lesions. Clin Orthop Relat Res 467:3307
3320, 2009.
2. Leontaritis N, Hinojosa L, Panchbhavik VK. Arthroscopically detected intra-
articular lesions associated with acute ankle fractures. J Bone Joint Surg Am
91:333339, 2009.
3. Elias I, Zoga AC, Morrison WB, Besser MP, Schweitzer ME, Raikin SM. Osteo-
chondral lesions of the talus: localization and morphologic data from 424 patients
using a novel anatomical grid scheme. Foot Ankle Int 28:154161, 2007.
4. Hannon CP, Smyth NA, Murawski CD, Savage-Elliott I, Deyer TW, Calder JD,
Kennedy JG. Osteochondral lesions of the talus: aspects of current management.
Bone Joint J 96-B:164171, 2014.
5. Canale ST, Belding RH. Osteochondral lesions of the talus. J Bone Joint Surg Am
62:97102, 1980.
6. Davis MW. Bilateral talar osteochondritis dissecans with lax ankle ligaments:
report of a case. J Bone Joint Surg Am 52:168170, 1970.
7. Delanois RE, Mont MA, Yoon TR, Mizell M, Hungerford DS. Atraumatic osteonec-
rosis of the talus. J Bone Joint Surg Am 80:529536, 1998.
8. Gribble PA, Hertel J, Plisky P. Using the star excursion balance test to assess
dynamic postural-control decits and outcomes in lower extremity injury: a
literature and systematic review. J Athl Train 47:339357, 2012.
9. Lunsford BR, Perry J. The standing heel-rise test for ankle plantar exion: criterion
for normal. Phys Ther 75:694698, 1995.
10. Sman AD, Hiller CE, Imer A, Ocsing A, Burns J, Refshauge KM. Design and reliability
of a novel heel rise test measuring device for plantarexion endurance. Biomed
Res Int 2014:17, 2014.
11. Berndt AL, Harty M. Transchondral fractures (osteochondritis dissecans) of the
talus. J Bone Joint Surg Am 41-A:9881020, 1959.
12. Hepple S, Winson IG, Glew D. Osteochondral lesions of the talus: a revised clas-
sication. Foot Ankle Int 20:789793, 1999.
13. Murphy JS, Mubarak SJ. Talocalcaneal coalitions. Foot Ankle Clin 20:681691, 2015.
14. Reilingh ML, Beimers L, Tuijthof GJ, Stufkens SA, Maas M, Van Dijk CN. Measuring
hindfoot alignment radiographically: the long axial view is more reliable than the
hindfoot alignment view. Skeletal Radiol 39:11031108, 2010.
15. Olney BW, Asher MA. Excision of symptomatic coalition of the middle facet of the
talocalcaneal joint. J Bone Joint Surg Am 69:539544, 1987.
16. Gantsoudes GD, Roocroft JH, Mubarak SJ. Treatment of talocalcaneal coalitions.
J Pediatr Orthop 32:301307, 2012.
17. Walther M, Kriegelstein S, Altenberger S, Ro ser A. Percutaneous calcaneal sliding
osteotomy. Oper Orthop Traumatol 28:309320, 2016.
18. Baumhauer JF, Nawoczenski DA, DiGiovanni BF, Wilding GE. Reliability and validity
of the American Orthopaedic Foot and Ankle Society clinical rating scale: a pilot
study for the hallux and lesser toes. Foot Ankle Int 27:10141019, 2006.
19. Maceira E, Monteagudo M. Subtalar anatomy and mechanics. Foot Ankle Clin
20:195221, 2015.
20. Frigg A, Nigg B, Hinz L, Valderrabano V, Russell I. Clinical relevance of hindfoot
alignment view in total ankle replacement. Foot Ankle Int 31:871879, 2010.
21. Paul J, Hinterwimmer S, Vavken P, Wo rtler K, Imhoff AB. Association between
hindfoot alignment and localisation of osteochondral lesions of the talus. Zeits-
chrift Orthop Unfallchir 152:389392, 2014.
22. Mosca VS, Bevan WP. Talocalcaneal tarsal coalitions and the calcaneal lengthening
osteotomy: the role of deformity correction. J Bone Joint Surg Am 94:15841594, 2012.
23. Hotel T, Engelhardt M. Osteochondrosis dissecans des Talus bei
Ru ckfumalalignmentdAutologe Matrixinduzierte Chondrogenese mit lateraler
Calcaneusverl angerungsosteotomie bei einer international erfolgreichen Nach-
wuchs-Skirennla uferin. Sport 29:118121, 2014.
24. Letts M, Davidson D, Ahmer A. Osteochondritis dissecans of the talus in children.
J Pediatr Orthop 23:617625, 2003.
25. Zanon G, Di Vico G, Marullo M. Osteochondritis dissecans of the talus. Joints
2:115123, 1978.
26. Raikin SM. Stage VI: massive osteochondral defects of the talus. Foot Ankle Clin
9:737744, 2004.
27. Raikin SM. Fresh osteochondral allografts for large-volume cystic osteochondral
defects of the talus. J Bone Joint Surg Am 91:28182826, 2009.
28. Haene R, Qamirani E, Story RA, Pinsker E, Daniels TR. Intermediate outcomes of
fresh talar osteochondral allografts for treatment of large osteochondral lesions of
the talus. J Bone Joint Surg Am 94:11051110, 2012.
29. Vannini F, Cavallo M, Baldassarri M, Castagnini F, Giannini S. Treatment of
juvenile osteochondritis dissecans of the talus: current concepts review. Joints
2:188191, 2014.
30. SooHoo NF. Comparison of reoperation rates following ankle arthrodesis and total
ankle arthroplasty. J Bone Joint Surg 89:21432149, 2007.
31. Jurina A, Dimnjakovi
c D, Mustapi
c M, Smoljanovi
c T, Bojani

c I. Clinical and MRI
outcomes after surgical treatment of osteochondral lesions of the talus in skele-
tally immature children. J Pediatr Orthop, 2016. http://dx.doi.org/10.1097/
BPO.0000000000000745 [Epub ahead of print].