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Arthrosc Tech
Arthrosc Tech
Arthroscopy Techniques
2212-6287
Elsevier

S2212-6287(24)00107-5
10.1016/j.eats.2024.103005
103005
Technical Note
Foot and Ankle
Arthroscopic Management of Dysplasia Epiphysealis Hemimelica (Trevor Disease) of the Ankle
Lui Tun Hing M.B.B.S. (HK), F.R.C.S. (Edin), F.H.K.A.M., F.H.K.C.O.S. luithderek@yahoo.co.uk
a∗
Lam Pui Ying Mb.Ch.B. b
a Department of Orthopaedics and Traumatology, North District Hospital, Hong Kong SAR, China
b Tuen Mun Hospital, Hong Kong SAR, China
∗ Address correspondence to Tun Hing Lui, M.B.B.S. (HK), F.R.C.S. (Edin), F.H.K.A.M., F.H.K.C.O.S., Department of Orthopaedics and Traumatology, North District Hospital, 9 Po Kin Road, Sheung Shui, NT, Hong Kong SAR, China. luithderek@yahoo.co.uk
30 4 2024
8 2024
30 4 2024
13 8 1030056 2 2024
13 3 2024
© 2024 The Authors
2024
https://creativecommons.org/licenses/by-nc-nd/4.0/ This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Dysplasia epiphysealis hemimelica (Trevor disease) is a rare skeletal development disorder of childhood, characterized by irregular ossification centers, which may develop together or individually, leading to asymmetric epiphyseal cartilage overgrowth, affecting 1 side of the epiphyses or the epiphyses equivalents (the medial side being affected twice as often as the lateral), until skeletal maturity is reached. Trevor disease around the ankle is locally aggressive with a poor outcome, especially in tumors involving the articular surface. The purpose of this Technical Note is to describe the details of arthroscopic management of dysplasia epiphysealis hemimelica (Trevor disease) of the ankle.

Technique Video

Video 1

Arthroscopic management of dysplasia epiphysealis hemimelica (Trevor disease) of the right ankle. The patient is in a supine position with a triangular supporting frame under the knee to keep the knee flexed. The anteromedial portal is the viewing portal, and the anterolateral portal is the working portal. Arthroscopic synovectomy of the anterolateral ankle is performed. The major protuberance is assessed and the portion causing anterior ankle impingement is resected. Then, the anterolateral ankle impingement is demonstrated and the portion of the protuberance anterior to the lateral malleolus is resected. The resultant chondral lesion at the talar body is microfractured. The arthroscope is switched to the anterolateral portal, and the other intra-articular pathologies are examined and treated accordingly.
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pmcDysplasia epiphysealis hemimelica (Trevor disease) is a rare skeletal development disorder of childhood, characterized by irregular ossification centers, which may develop together or individually, leading to asymmetric cartilage overgrowth of 1 side of the epiphyses/epiphyses equivalents, until skeletal maturity is reached.1, 2, 3, 4, 5, 6 It is a nonhereditary disorder, and no environmental determinant is identified.1, 2, 3,5,6 It most commonly affects children in the 2- to 14-year age group and has a 3 times higher male preponderance.1,7,8 Trevor disease can be classified as “localized” (only 1 epiphysis is affected), “classic” (more than 1 epiphysis in the same limb is affected), or “generalized” (the entire limb is affected).1,8 The histopathologic diagnosis is not pathognomonic as it resembles osteochondroma, with varying degrees of endochondral ossification as well as hyaline cartilage cap.1, 2, 3,8,9 EXT1 and EXT2 gene expression studies differentiate Trevor disease (normal gene expression) from osteochondroma (low gene expression).2,7, 8, 9 Because these are expensive, clinical and radiologic findings are important for establishing a diagnosis.2,9 The characteristic radiographic findings include asymmetrical epiphyseal overgrowth of osteochondroma-like appearance.5,6 The lower limb is most frequently affected, especially the ankle joint, followed by the knee joint.1,2,8 Trevor disease around the ankle can be locally aggressive, with instability, deformity, growth disturbance, and osteoarthritis.1

Treatment of Trevor disease depends on symptoms, severity of the disease, and location of the lesion. It may vary from clinical monitoring to surgery, including complete or incomplete resection of the lesion, corrective osteotomies, and hemi-epiphysiodesis.4,6,9, 10, 11 Asymptomatic patients are treated nonoperatively, under close follow-up, until skeletal maturity. Operative management is reserved for symptomatic lesions. If the lesion causes epiphyseal asymmetry or mechanical block of the ankle motion, resection of the exostosis is required.7 The surgical goal is complete removal of the osseous growth, without injuring the epiphysis.1 This is feasible for extra-articular or juxta-articular lesions but may not be for intra-articular lesions.1,6 Intra-articular lesions tend to be complicated with recurrence and deformities. Lesion excision may induce hyperemia and further abnormal bone growth.12 Excision of intra-articular lesions may lead to secondary degenerative changes.12

The arthroscopic approach should be considered for patients with intra-articular lesions.3,7,13, 14, 15 The purpose of this Technical Note is to describe the details of arthroscopic management of dysplasia epiphysealis hemimelica of the ankle. It is indicated for symptomatic dysplasia epiphysealis hemimelica of the ankle with major protuberance at the lateral part of the talar body causing anterior ankle impingement. It is contraindicated for dysplasia epiphysealis hemimelica of the ankle with posterior ankle impingement or the lesion is at the medial part of the talar body (Table 1).Table 1 Indications and Contraindications of Arthroscopic Management of Dysplasia Epiphysealis Hemimelica (Trevor Disease) of the Ankle

Indications	Contraindications	
1. Symptomatic dysplasia epiphysealis hemimelica of the ankle with major protuberance at the lateral part of the talar body causing anterior ankle impingement

	1. Dysplasia epiphysealis hemimelica of the ankle with posterior ankle impingement

2. The lesion of the dysplasia epiphysealis hemimelica is at the medial part of the talar body

	

Technique

Preoperative Assessment and Patient Positioning

The pain and tenderness should be located carefully. Any limitation of ankle motion or ankle/hindfoot deformity is noted. Radiographs show a partially ossified, lobulated osteochondral protuberance arising unilaterally from the affected epiphysis with or without an osseous connection.1,2 Lateral radiographs with the ankle dorsiflexed and plantarflexed may demonstrate any mechanical block of ankle motion (Fig 1). In this illustrated case, the major protuberance is mainly at the lateral talar body with secondary deformity of the tibial plafond and distal tibiofibular syndesmosis. A computed tomography (CT) scan can help to differentiate the pathology from the normal physis and is useful for planning resection (Fig 2).1 A 3-dimensional CT scan can provide an overall picture of the lesion for preoperative planning and parents’ understanding of the disease (Fig 3).6 Magnetic resonance (MR) imaging helps to delineate the soft tissue margins and the extent of epiphyseal involvement, as well as differentiate the epiphyseal tissue from the accessory ossification center (Fig 4).1 Sometimes, scintigraphy can be used to detect multiple lesions at a much lower radiation dose than a skeletal survey.2Fig 1 Arthroscopic management of dysplasia epiphysealis hemimelica (Trevor disease) of the right ankle. The patient is in a supine position with a triangular supporting frame under the knee to keep the knee flexed. Radiographs of the illustrated case’s right ankle. (A) Anteroposterior view shows the major protuberance is at the lateral side of the talar dome with corresponding deformation of the tibial plafond. (B) Lateral view with the ankle plantarflexed. (C) Lateral view with the ankle dorsiflexed. (P, protuberance.)

Fig 2 Arthroscopic management of dysplasia epiphysealis hemimelica (Trevor disease) of the right ankle. The patient is in a supine position with a triangular supporting frame under the knee to keep the knee flexed. Computed tomography scan of the illustrated case. (A) Coronal view shows that beside the major protuberance at the lateral talar dome, there is another small protuberance at the central part of the talar dome. (B) Sagittal view shows multiple ossifications of the protuberance. (P, protuberance.)

Fig 3 Arthroscopic management of dysplasia epiphysealis hemimelica (Trevor disease) of the right ankle. The patient is in a supine position with a triangular supporting frame under the knee to keep the knee flexed. Three-dimensional computed tomography scan of the illustrated case. (A) Lateral view of the ankle. (B) Posterior view of the ankle. (C) Anterior view of the ankle. Panels A, B, and C demonstrate the relationship of the major protuberance to the lateral malleolus. (D) Anterolateral view of the talus. (E) Anteromedial view of the talus. Panels D and E clearly show the anatomy of the protuberance. (P, protuberance.)

Fig 4 Arthroscopic management of dysplasia epiphysealis hemimelica (Trevor disease) of the right ankle. The patient is in a supine position with a triangular supporting frame under the knee to keep the knee flexed. Magnetic resonance imaging of the illustrated case. (A) Coronal view shows that the major protuberance is at the lateral side of the talar dome with corresponding deformation of the tibial plafond. (B) Sagittal view shows the protuberance anterior to the lateral malleolus. (C) Sagittal view shows multiple ossifications of the major protuberance. (D) Sagittal view shows a small protuberance at the central part of the talar dome. (P, protuberance.)

The patient is in a supine position with a triangular supporting frame (Innomed) under the knee to keep the knee flexed. A thigh tourniquet is applied to provide a bloodless operative field. A 2.7-mm, 30° arthroscope (Henke Sass Wolf GmbH) is used for this procedure. Fluid inflow is driven by gravity and no fluid inflow is required.

Before the arthroscopic surgery, intraoperative fluoroscopic examination of the ankle is performed, and in this illustrated case, the mechanical block of ankle dorsiflexion by the protuberance is confirmed (Fig 5).Fig 5 Arthroscopic management of dysplasia epiphysealis hemimelica (Trevor disease) of the right ankle. The patient is in a supine position with a triangular supporting frame under the knee to keep the knee flexed. Intraoperative fluoroscopy of the illustrated case. Ankle dorsiflexion is limited by mechanical block by the major protuberance. (A) Anteroposterior view. (B) Lateral view with the ankle maximally plantarflexed. (C) Lateral view with the ankle maximally dorsiflexed. (P, protuberance.)

Portal Placement

Ankle arthroscopy is performed via the anteromedial and anterolateral portals at the anterior ankle joint line. The anterolateral portal is at the lateral side of the peroneus tertius tendon. The anteromedial portal is at the lateral side of the tibialis anterior tendon, which can improve the visualization of the lateral ankle gutter via this portal (Fig 6).Fig 6 Arthroscopic management of dysplasia epiphysealis hemimelica (Trevor disease) of the right ankle. The patient is in a supine position with a triangular supporting frame under the knee to keep the knee flexed. Ankle arthroscopy is performed via the anteromedial and anterolateral portals at the anterior ankle joint line. The anterolateral portal is at the lateral side of the peroneus tertius tendon. The anteromedial portal is at the lateral side of the tibialis anterior tendon, which can improve the visualization of the lateral ankle gutter via this portal. (ALP, anterolateral portal; AMP, anteromedial portal.)

Synovectomy of Anterolateral Ankle

The anteromedial portal is the viewing portal, and the anterolateral portal is the working portal. Arthroscopic synovectomy of the anterolateral ankle is performed with an arthroscopic shaver (Dyonics; Smith & Nephew) (Fig 7).Fig 7 Arthroscopic management of dysplasia epiphysealis hemimelica (Trevor disease) of the right ankle. The patient is in a supine position with a triangular supporting frame under the knee to keep the knee flexed. The anteromedial portal is the viewing portal, and the anterolateral portal is the working portal. Arthroscopic synovectomy of the anterolateral ankle is performed with an arthroscopic shaver. (AS, arthroscopic shaver; IS, inflamed synovium; P, protuberance; TD, talar dome.)

Assessment of the Talar Protuberance

The anteromedial portal is the viewing portal, and the anterolateral portal is the working portal. The position and extent of the talar protuberance are assessed. The corresponding articular deformity at the tibial plafond is noted (Fig 8).Fig 8 Arthroscopic management of dysplasia epiphysealis hemimelica (Trevor disease) of the right ankle. The patient is in a supine position with a triangular supporting frame under the knee to keep the knee flexed. The anteromedial portal is the viewing portal, and the anterolateral portal is the working portal. The position and extent of the talar protuberance are assessed. The corresponding articular deformity at the tibial plafond is noted. (P, protuberance; T, tibia; TD, talar dome.)

Assessment of the Portion of the Protuberance Causing Anterior Ankle Impingement

The anteromedial portal is the viewing portal, and the anterolateral portal is the working portal. The ankle is dorsiflexed, and the anterior portion of the protuberance causing anterior ankle impingement is noted (Fig 9).Fig 9 Arthroscopic management of dysplasia epiphysealis hemimelica (Trevor disease) of the right ankle. The patient is in a supine position with a triangular supporting frame under the knee to keep the knee flexed. The anteromedial portal is the viewing portal, and the anterolateral portal is the working portal. The ankle is dorsiflexed and the anterior portion of the protuberance causing anterior ankle impingement is noted. (P, protuberance; T, tibia.)

Resection of the Anterior Protuberance Causing Ankle Impingement

The anteromedial portal is the viewing portal, and the anterolateral portal is the working portal. The anterior portion of the protuberance causing anterior ankle impingement is detached from the talar body by means of an arthroscopic shaver, an arthroscopic probe (Acufex; Smith & Nephew), and an arthroscopic curette (Acufex; Smith & Nephew) through the cleavage plane between the protuberance and the talar body (Fig 10). The detached protuberance is removed by a hemostat.Fig 10 Arthroscopic management of dysplasia epiphysealis hemimelica (Trevor disease) of the right ankle. The patient is in a supine position with a triangular supporting frame under the knee to keep the knee flexed. The anteromedial portal is the viewing portal, and the anterolateral portal is the working portal. (A) The anterior portion of the protuberance causing anterior ankle impingement is detached from the talar body by means of an arthroscopic shaver through the cleavage plane between the protuberance and the talar body. (B) The fragment is further detached from the talar body by means of an arthroscopic curette. (P, protuberance; RC, ring curette; TD: talar dome.)

Assessment of the Portion of the Protuberance Causing Anterolateral Ankle Impingement

The anteromedial portal is the viewing portal, and the anterolateral portal is the working portal. Residual limitation of ankle dorsiflexion is due to anterolateral impingement between the lateral malleolus and the protuberance anterior to the lateral malleolus. The majority of the lateral malleolus is not visible via the anteromedial portal. The scope can be shifted plantarly to see the lateral malleolar tip. The anterolateral impingement can be confirmed by the arthroscopic probe with the ankle dorsiflexed (Fig 11).Fig 11 Arthroscopic management of dysplasia epiphysealis hemimelica (Trevor disease) of the right ankle. The patient is in a supine position with a triangular supporting frame under the knee to keep the knee flexed. The anteromedial portal is the viewing portal, and the anterolateral portal is the working portal. The ankle dorsiflexion range is assessed again, and the residual limitation is due to anterolateral impingement between the lateral malleolus and the protuberance anterior to the lateral malleolus. The majority of the lateral malleolus is not visible via the anteromedial portal. The scope can be positioned plantarly to see the lateral malleolar tip. The anterolateral impingement can be confirmed by the arthroscopic probe with the ankle dorsiflexed. (AP, arthroscopic probe; LM, lateral malleolus; P, protuberance.)

Resection of the Lateral Portion of Protuberance Anterior to the Lateral Malleolus

The anteromedial portal is the viewing portal, and the anterolateral portal is the working portal. The cleavage plane between the prominence and the talar body is dissected by the arthroscopic probe. The portion of the protuberance merged with the talar body is osteotomized from the cleavage plane with an arthroscopic osteotome (Acufex; Smith & Nephew). After removal of the protuberance anterior to the lateral malleolus, the whole lateral malleolus is exposed (Fig 12). The ankle dorsiflexion motion is confirmed to be restored.Fig 12 Arthroscopic management of dysplasia epiphysealis hemimelica (Trevor disease) of the right ankle. The patient is in a supine position with a triangular supporting frame under the knee to keep the knee flexed. The anteromedial portal is the viewing portal, and the anterolateral portal is the working portal. The cleavage plane between the prominence and the talar body is carefully dissected by the arthroscopic probe. (A) The portion of the protuberance merged with the talar body is osteotomized from the cleavage plane. (B) After removal of the protuberance anterior to the lateral malleolus, the whole lateral malleolus is exposed. (AO, arthroscopic osteotome; LM, lateral malleolus; P, protuberance; TD, talar dome.)

Microfracture of the Chondral Defect

The anteromedial portal is the viewing portal, and the anterolateral portal is the working portal. After removal of the protuberance fragment, the resultant chondral defect at the talar dome is microfractured with an arthroscopic awl (Smith & Nephew) (Fig 13).Fig 13 Arthroscopic management of dysplasia epiphysealis hemimelica (Trevor disease) of the right ankle. The patient is in a supine position with a triangular supporting frame under the knee to keep the knee flexed. (A) The protuberance fragments removed. (B) The anteromedial portal is the viewing portal, and the anterolateral portal is the working portal. After removal of the protuberance fragment, the resultant chondral defect at the talar dome is microfractured with an arthroscopic awl. (AA, arthroscopic awl; CD, chondral defect.)

Assessment of Other Intra-articular Pathologies

The anterolateral portal is the viewing portal, and the anteromedial portal is the working portal. The intra-articular pathologies of the other part of the ankle joint are assessed. In this illustrated case, the inflamed synovium of the anteromedial and posteromedial ankle is resected. The chondral budding at the medial talar facet is resected. Tramline chondral lesion is noted at the tibial plafond (Fig 14, Table 2, Video 1).Fig 14 Arthroscopic management of dysplasia epiphysealis hemimelica (Trevor disease) of the right ankle. The patient is in a supine position with a triangular supporting frame under the knee to keep the knee flexed. The anterolateral portal is the viewing portal, and the anteromedial portal is the working portal. The intra-articular pathologies of the other part of the ankle joint are assessed. (A) Inflamed synovium of the anteromedial ankle. (B) Inflamed synovium of the posteromedial ankle. (C) Chondral budding at the medial talar facet. (D) Tramline chondral lesion at the tibial plafond. (AS, arthroscopic shaver; CB, chondral budding; IS, inflamed synovium; MM, medial malleolus; TD, talar dome; TL, tramline chondral lesion of the talar plafond; Tmf, medial facet of talar body.)

Table 2 Pearls and Pitfalls of Arthroscopic Management of Dysplasia Epiphysealis Hemimelica (Trevor Disease) of the Ankle

Pearls	Pitfalls	
1. Careful surgical planning by clinical assessment and imaging is the key to success.

2. Identification of the cleavage plane between the lesion and articular surface can avoid damage to the normal articular cartilage.

3. Resection of the inflamed synovium behind the lesion is important for pain control.

4. The anterolateral ankle impingement can be assessed at the level of the lateral malleolar tip.

	1. Excising the intra-articular lesion with compensatory deformity of the tibial plafond and secondary joint congruity will lead to iatrogenic joint incongruity.

	

Discussion

Trevor disease around the ankle is locally aggressive with a poor outcome, especially in tumors involving the articular surface.16 Early removal of localized intra-articular lesions should be considered, even asymptomatic, to improve the joint congruity and avoid the risk of articular cartilage damage and early osteoarthritis.5, 6, 7, 8,15

A cleavage plane exists between this lesion and the epiphysis before they coalesce and eventually fuse.4 MR imaging is useful to find this cleavage plane.5 Early removal of the intra-articular lesions has the advantage that the cleavage plane is still identifiable, and irreversible joint damage and deformity have not yet occurred.5,8 Resection of masses attached to the articular cartilage at the late stage may jeopardize the remaining joint surface because differentiating the lesion from normal cartilage is not often possible.10 If the joint has integrated the intra-articular lesion by deformation of the opposing articular surface, the lesion should be left untouched.6 Arthroscopic resection of the intra-articular lesions can avoid damaging the healthy articular cartilage and remove only the protruding masses that are blocking joint motion.

The advantages of this technique include small incisions and better cosmetic outcome, minimal soft tissue trauma, accurate assessment of the lesions, and detection of other intra-articular pathologies. The potential risks of this technique include injury to the branches of the superficial peroneal nerve, deep peroneal nerve, extensor tendons, and articular cartilage (Table 3). This procedure is technically difficult and should be reserved for experienced ankle arthroscopists.Table 3 Advantages and Risks of Arthroscopic Management of Dysplasia Epiphysealis Hemimelica (Trevor Disease) of the Ankle

Advantages	Risks	
1. Small incisions and better cosmetic outcome

2. Minimal soft tissue trauma

3. Accurate assessment of the lesions

4. Detection of other intra-articular pathologies

	1. Injury to the branches of the superficial peroneal nerve

2. Injury to the deep peroneal nerve

3. Injury to the extensor tendons

4. Injury to the articular cartilage

	

Disclosures

All authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.

Supplementary Data

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Video 1

Arthroscopic management of dysplasia epiphysealis hemimelica (Trevor disease) of the right ankle. The patient is in a supine position with a triangular supporting frame under the knee to keep the knee flexed. The anteromedial portal is the viewing portal, and the anterolateral portal is the working portal. Arthroscopic synovectomy of the anterolateral ankle is performed. The major protuberance is assessed and the portion causing anterior ankle impingement is resected. Then, the anterolateral ankle impingement is demonstrated and the portion of the protuberance anterior to the lateral malleolus is resected. The resultant chondral lesion at the talar body is microfractured. The arthroscope is switched to the anterolateral portal, and the other intra-articular pathologies are examined and treated accordingly.
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References

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