Vascular Complications Caused by Tibial Osteochondroma: History
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Andrea Angelini
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Osteochondromas are the most common benign primary bone tumors in growing patients representing about 35–45% of all benign bone tumors. About 90% of these occur sporadically and are usually solitary. Osteochondromas occur most often around the knee (40%) and proximal tibial is affected in 15–20%. Vascular complications are rare and include vessel perforation and thrombosis, arterial thromboembolic events and pseudoaneurysm formation. 

  • osteochondroma
  • treatment

1. Introduction

Osteochondromas are common benign tumors of bone, most frequently found in adolescents or young adults. Osteochondromas can be sessile or pedunculated, presenting as a solitary lesion in 85%, while 15% occur in the context of hereditary multiple exostoses (HME), a genetic autosomal dominant disorder. Although often asymptomatic and discovered incidentally, symptoms may derive from compression or dislocation of near structures such as adjacent vessels or nerves, fractures, osseous deformities, bursa formation or malignant transformation [1]. Cartilage cap thickness > 2 cm in adults or >3 cm in children as well as new onset of pain or growth, or rapid growth of the lesion, especially after the closure of the growth plate, might reflect cancerous transformation [2]. The involvement of lower extremities is common, particularly metaphyseal structures of the femur and tibia around the knee joint [3][4]. Surgical resection is indicated for symptomatic lesions, complications, cosmetic reasons or malignant transformation. Local recurrence is less than 2% if complete resection is achieved.
Vascular complications are rare and include vessel perforation and thrombosis, arterial thromboembolic events and pseudoaneurysm formation [3]. From an epidemiologic point of view, pseudoaneurysms are most frequently observed compared to thromboembolic complications [5]. The pathogenesis and development of these vascular complications are unknown because the exostoses are usually not detected until the secondary symptoms appear. Only few with osteochondromas of the proximal tibial causing popliteal artery thrombosis and acute lower limb ischemia have been reported [6][7][8]. It is to underline the importance of a combined vascular-orthopedic approach to correct diagnosis and prompt surgical management of vascular complications caused by tibial osteochondromas.

2. Diagnosis

Pedunculated osteochondromas around the knee have a typical appearance, extending away from the adjacent joint, described as stalactites and stalagmites. In any bone that develops from enchondral ossification, osteochondromas have a marrow, cortical and periosteum continuity with the underlying native bone. This direct continuity clearly appreciable in CT scan is a pathognomonic finding [9]. The cartilaginous cup is the most important area that should be investigated. On MRI, the cartilaginous cap is typically hypointense on T1-weighted images and variable on T2 images (hypointense or hyperintense if densely or poorly mineralized, respectively) [9]. Angiography and other vascular studies should be considered in the presence of vascular compression or for surgical planning [7][10][11][12]. Sometimes these exams are helpful to characterize neovascularity, which may signal malignant transformation [7][11].
It is important to include bone tumors in differential diagnosis in patients with vascular symptoms. The adequate approach for these patients includes clinical evaluation, plain radiographs, CT scan and MRI. In the presence of bone tumors, CT and MRI are the most sensitive examinations to exclude malignant appearance, soft tissue extension and signs of popliteal entrapment syndrome (PES). In young adult patients presenting with intermittent claudication or vascular insufficiency mimicking atherosclerotic peripheral vascular disease, alternative causes such as trauma, tumors, thromboangioitis obliterans, fibromuscular dysplasia, vasculitis and PES should be suspected [13]. PES refers to a group of symptoms caused by mechanical compression of popliteal artery, vein or tibial nerve in the popliteal fossa. This is usually related to musculotendinous structures, but a few was described bony exostoses of the popliteal fossa causing PES [3][6]. Angiography and ultrasound are useful to define the precise level of vascular defects. In patients with HME, popliteal pseudoaneurysms is more frequently diagnosed in the context of a non–traumatic event (65%) than after trauma (35%), and symptoms consist of pain and swelling on the popliteal fossa with preserved distal blood flow [14]. Nevertheless, the presence of ecchymosis or pulsatile mass in the popliteal region is not frequently observed but is highly suggestive of a popliteal pseudoaneurysm [14]. Duplex ultrasound is the gold of standard for diagnosis of pseudoaneurysms because it can clearly depict the vascular lesion and its relationship with the osteochondroma [15]. However, duplex ultrasound has some limitations in patients with obesity or huge hematomas due to more thickness between the vascular structures and the transductor. In these, it has been argued that in favour of angiography to depict the anatomic details before surgery. [16].
With the reported one, as underlined by other, it was strongly supported that the use of doppler ultrasound because it could be used as a functional examination showing the blood flow asset during complete extension of the knee [1][3][13][17][18][19][20]. Given the benign nature of osteochondromas, surgical resection is considered the gold standard of treatment, however it was suggested a combined vascular-orthopedic approach from the onset of symptoms and diagnosis [21]. When surgery is planned, a complete excision of the cartilaginous cap is critical to prevent recurrence.

3. Treatment

The main consideration about management is how to approach the vascular complication considering that treatment of osteochondroma is mainly simple surgical resection at the base of the tumor with meticulous dissection of the neurovascular structures. Most were managed by osteochondroma resection with or without open vascular reconstruction. Open surgical options include bypass and interposition grafting, direct repair, patch angioplasty, and aneurysm resection with direct end-to-end anastomosis. Arterial reconstruction should be considered by either closing the arterial defect in cases of pseudoaneurysm formation or using a saphenous vein graft for arterial thrombosis and extensive arterial damage. From this, pseudoaneurysm resection associated with venous bypass or end-to-end anastomosis represent the most frequent surgical approach, because of the high risk of recurrent pseudoaneurysm or stenosis after direct closure. Vasseur et al. reviewed 97 patients with osteochondroma associated with vascular complications, surgically repaired [11]. Among them, direct closure was performed in 25 with a higher risk of recurrent pseudoaneurysm if stress is applied to the suture line (as the patient ages). Moreover, revascularization is considered the gold standard surgical option for preventing tissue damage or limb loss from embolic events in the setting of acute limb ischemia [3]. Most of the surgeons suggest a complete removal of the damaged wall and the use of patch or saphenous vein for defect repair, to reduce the rate of late complications.
Endovascular management is infrequently described. In two hybrid procedures, one patient underwent thrombectomy for embolus in the tibioperoneal trunk with subsequent vein patching of the popliteal pseudoaneurysm, and another had coil embolization of the pseudoaneurysm followed by direct repair of the artery one week later [19][22]. Sometimes, treatment of vascular complication consists of emergency surgery. Banno et al. reported a pseudoaneurysm of the popliteal artery in a 16-year-old boy with HMO [23]. Duarte at al. recently reported a 15-year-old patient with a history of current massages as part of his gym routine, who arrived at the emergency department with four days of pain, and ecchymosis in the right popliteal region: duplex ultrasonography and arteriography confirming the diagnosis of popliteal pseudoaneurysm [15]. Nasr at al. described vascular complications that occurred in a solitary form and the other with HMO, for a lower limb ischemia and pseudoaneurysm of the left superficial femoral artery [13]. Chen at al. reported that 130 vascular complications from osteochondroma in all sites. Vascular complications were due to osteochondromas of the femur (66.2%), tibia (15.4%), fibula (7.7%), humerus (5.4%), clavicle (1.5%), ribs (1.5%), pubic ramus (0.8%), scapula (0.8%) and cervical vertebra (0.8%). The popliteal artery was involved in 66%, and popliteal pseudoaneurysms were the most prevalent vascular complication (49%) [24]. For 38-year-old man was reported, presenting with left calf pain and swelling due to a popliteal pseudoaneurysm and incidental peroneal vein thrombosis secondary to a fractured femoral sessile osteochondroma, treated with resection of the osteochondroma, excision of the aneurysm, and primary end-to-end anastomosis of the artery [24].
Local recurrence of osteochondroma after resection is low at 1.8% but may be higher in skeletally immature patients [24][25]. Two of delayed pseudoaneurysm formation after incomplete osteochondroma resection have been reported. In one a partially resected osteochondroma resulted in pseudoaneurysm formation five years later because of ongoing arterial trauma from the residual tumor’s irregular edges [21]. Scotti et al. reported brachial pseudoaneurysm presenting with rupture secondary to a partially resected osteochondroma three years earlier [26]. Combined vascular-orthopedic approach was initiated with intra-arterial locoregional thrombolytic therapy in place for four days and for the surgical tangential resection of the tibial osteochondroma.

This entry is adapted from the peer-reviewed paper 10.3390/diagnostics12051191

References

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