|Year : 2023 | Volume
| Issue : 2 | Page : 164-168
Management of Buerger's disease (thromboangiitis obliterans) of the lower limb by horizontal distraction and corticotomy by Ilizarov's technique
Kumar Rohit, Vikas Verma
Department of Orthopedics, Mahatma Gandhi University of Medical Science and Technology, Jaipur, Rajasthan, India
|Date of Submission||22-Oct-2022|
|Date of Decision||07-Dec-2022|
|Date of Acceptance||12-Dec-2022|
|Date of Web Publication||3-May-2023|
Department of Orthopedics, Mahatma Gandhi University of Medical Science and Technology, Sitapura, Jaipur - 302 022, Rajasthan
Source of Support: None, Conflict of Interest: None
Introduction: Thromboangiitis obliterans (TAO), also known as Buerger's disease, is a disease of unknown etiology affecting small- and medium-sized blood vessels, mainly seen in heavy smokers. The characteristic features of this disease are distal ischemia, with patients having claudication, rest pain, and toe ulcers. The proper management of TAO is still a challenging problem. Various medical and surgical treatments have been tried with a variable success. Since it is a disease characterized by chronic ischemia, achieving neoangiogenesis could have a beneficial effect. Ilizarov has shown that corticotomy and distraction of bony fragments can increase the blood supply to the entire limb. TAO can be managed using this principle of distraction osteogenesis to induce neoangiogenesis. We conducted this study to evaluate the clinical outcomes of longitudinal tibial corticotomy and horizontal distraction with Ilizarov's fixator in Buerger's disease of the lower limb. Materials and Methods: Ten patients with clinically and radiologically proven Buerger's disease admitted to our hospital between 2019 and 2021 who had failed the conservative pharmacological modalities of treatment were included. A lateral tibial corticotomy and distraction was performed by the use of olive wires and two-ring frame with horizontal distraction mechanism. Lateral distraction was started after 10 days at the rate of 0.25 mm 6 h for 25 days to achieve a distraction of 2.5 cm. The frame was removed after consolidation of regenerate. Results: Of the 10 cases, eight patients became pain free, while one had a partial relief of pain and one patient worsened on treatment and underwent below-knee amputation. Three patients had at least one episode of pin site infection which improved with oral antibiotics and dressings. One patient had delayed consolidation of regenerate. The claudication distance improved at each follow-up for all but one patient. Conclusion: Neoangiogenesis by corticotomy and distraction offers a relatively simple and cost-effective surgical option to salvage ischemic limbs from amputations and provide pain relief, ischemic ulcer healing, and improved claudication distance, thus improving the quality of life.
Keywords: Amputation, Buerger's disease, distraction osteogenesis, Ilizarov ring fixator, thromboangiitis obliterans
|How to cite this article:|
Rohit K, Verma V. Management of Buerger's disease (thromboangiitis obliterans) of the lower limb by horizontal distraction and corticotomy by Ilizarov's technique. J Orthop Dis Traumatol 2023;6:164-8
|How to cite this URL:|
Rohit K, Verma V. Management of Buerger's disease (thromboangiitis obliterans) of the lower limb by horizontal distraction and corticotomy by Ilizarov's technique. J Orthop Dis Traumatol [serial online] 2023 [cited 2023 Jun 4];6:164-8. Available from: https://jodt.org/text.asp?2023/6/2/164/375542
| Introduction|| |
Thromboangiitis obliterans (TAO) is a disease of unknown etiology affecting small- and medium-sized blood vessels. It was first described in by Winiwarter who called it endarteritis. In 1908, Buerger called the syndrome TAO because microscopic features were characterized by thrombosis in both arteries and veins with a marked inflammatory response. Later, the condition became to be known as Buerger's disease., The characteristic features of this disease are distal ischemia, with patients having claudication, rest pain, and toe ulcers. TAO is more common in males and affects the younger age group of 20–40 years. Buerger's disease has a strong association with tobacco smoking. Discontinuation of tobacco use, therefore, is the first and most effective step.,,,,
The proper management of TAO is still a challenging problem. Various medical and surgical treatments have been tried with a variable success. Medical treatment with drugs, namely, iloprost, clofibrate, cyclophosphamide, and calcium channel blockers has been tried and shown to provide variable degree of improvement in outcome. Surgical modalities which have been attempted for treating Buerger's disease include thrombolytic therapy, arterial reconstruction, bypass vein grafts, lumbar sympathectomy, and microvascular omental transfer.,,, Recently, some procedures showing some promises are spinal cord stimulation, angiogenesis by autologous bone marrow mononuclear cell implantation, and gene therapy.,,, The ideal modality for treatment, however, is still undecided.
Since it is a disease characterized by chronic ischemia, achieving neoangiogenesis could have a beneficial effect. In 1951, Ilizarov has shown that corticotomy and distraction of bony fragments can increase the blood supply to the entire limb. The formation of new blood vessels after distraction takes place not only in the bones but also in the soft tissues equally., He was the first to use distraction osteogenesis in the management of TAO. In the treatment of TAO, bone widening stimulates considerable vascular hypertrophy in the regenerate, without altering the limb length. Since then, a few other studies have been attempted to study the effects of distraction for the treatment of TAO.,,,
We conducted this study to evaluate the clinical outcomes of longitudinal tibial corticotomy and horizontal distraction with Ilizarov's fixator in Buerger's disease of the lower limb.
| Materials and Methods|| |
Between 2019 and 2021, ten patients of clinically and radiologically proven Buerger's disease as per Shionoya's criteria of age >25 years who consented for the study were included. Patients having uncontrolled diabetes, severe infection, or absent femoral pulse were not included in the study. All the study participants were treated by horizontal distraction with Ilizarov's fixator at our hospital.
A thorough history and clinical examination were done for each patient. Color Doppler study was done to establish the diagnosis and assess the status of the distal vasculature. Computed tomography angiogram was not routinely done. The history of smoking was taken and documented as a number of pack years. The history of tobacco use in other forms was also taken. Clinical examination was done, and capillary refill time was noted. Palpation of femoral, dorsalis pedis, and posterior tibial arteries on both sides was done, and their presence or absence was documented. The claudication distance was measured and noted. If a gangrene or ulcer was present, it was also examined and treated.
All the patients were operated under spinal anesthesia on a standard operating table. No tourniquet was used. A two-ring Ilizarov frame consisting of two full rings was joined by the connecting rods anteromedially and posteromedially, and a horizontal distraction assembly on the lateral side was preconstructed according to the leg length.
Five small 0.8 cm –1 cm anterior incisions at 3 cm intervals were given starting 3 cm distal to the tibial tuberosity [Figure 1]a. The periosteum on the medial tibial surface was incised and elevated. Multiple drill holes were made with 2.7 mm drill bit on the medial surface of the tibia, 1 cm medial to the tibial crest aiming in the posterolateral direction so as they exit about 2 cm below the level of crest from the lateral surface [Figure 1]b. The corticotomy was then completed with a 5mm osteotome by joining the holes on the medial and lateral surfaces of tibia, the total longitudinal length of corticotomy being 12 cm. After completing the cuts, the corticotomy was pried open. The frame was then mounted by fixing the proximal ring at the level of head of the fibula and the distal ring at around 2 cm proximal to the ankle joint. The rings were fixed with Ilizarov wires and Schanz screws, following the standard Ilizarov techniques and safe zones. Finally, three parallel olive wires were passed through the corticotomy gap [Figure 1]c and [Figure 1]d anteromedially into the cut segment of the tibia directed in the posterolateral direction [Figure 2]. The olive wires were then attached with the distraction assembly attached them with the slotted bolts. Any ulcers and gangrenous toes if present were debrided and dressed. Gangrenous toes were not amputated at the time of index surgery. Amputation was done at follow-up if no healing was seen after starting distraction.
|Figure 1: Intraoperative images showing incisions (a), C-arm views showing corticotomy and olive wire placement (b), (c) and image of the limb after frame assembly (d)|
Click here to view
|Figure 2: Schematic diagram of tibial cross section showing direction of drilling (a), corticotomy (b), opening of cortex by prying (c) and olive wire insertion (d)|
Click here to view
Weight bearing as tolerated was started from the second postoperative day with the support of walker. Lateral distraction was started after 10 days at the rate of 0.25 mm 6 h for 25 days to achieve a distraction of 2.5 cm. The patients were reviewed in the outpatient clinic every 10 days till the completion of distraction. Stitches were removed at 2 weeks. X-rays were taken on each visit to ascertain proper distraction and to monitor formation and healing of the regenerate. Once regenerate seemed consolidated, frame was removed [Figure 3] and [Figure 4].
|Figure 3: Preoperative (a) and follow-up (b) Clinical images showing healing of toe gangrene|
Click here to view
|Figure 4: The final X-ray after the frame removal showing distraction and consolidation of regenerate|
Click here to view
Clinical examination at each visit was done to assess and record the healing time of ischemic ulcers. The severity of rest pain on Visual Analog Scale (VAS) and claudication distance were documented at 3 months, 6 months, and 1 year after surgery.
| Results|| |
A total of ten patients of Buerger's disease treated by the above-mentioned technique were included in our study. The mean age of patients was 42 years (range: 34–70 years). Nine of them were male and only one was female. Seven patients had right-side involvement, and three had left side involved. All patients had a history of long-standing tobacco smoking in the form of bidis or cigarettes, with the mean duration of smoking of 20 years. None of the patients had tried any surgical intervention earlier for this disease in our study. Four patients had gangrenous changes in one or more toes, which were not amputated at the time of surgery. In two of these patients, gangrenous wound was healed and amputation was thus avoided [Figure 3]. Eight patients had nonhealing ulcers over toes or planar aspect of the foot, which healed after surgery. No patient required skin graft or flap for coverage of ulcers or amputation wound.
The most frequent complication was superficial pin site infection. A total of three patients had at least one episode of pin site infection which improved with oral antibiotics and dressings. One patient had delayed consolidation of regenerate. This was managed by keeping the frame for 6 months till consolidation occurred. There was no relapse of disease or recurrence of symptoms.
The mean preoperative VAS pain score was 10, with all patients reporting severe pain even on rest. After surgery, there was a significant improvement in the VAS scores with the mean score of 2 at 3-month follow-up, 0.5 at 6-month follow-up, and 0.3 at 1-year follow-up.
The claudication distance at 3 months improved to >2 km in six patients, 1-2 km in two patients, and <1 km in two patients. It further improved at 6-month follow-up with eight patients reporting a claudication distance of >2 km. At 1 year also, only two patients reported a claudication distance of <1 km; however, one had an improvement in rest pain [Table 1].
Based on the grading system by Patwa et al., Six patients had an excellent outcome, two had a good outcome, one had a fair outcome, and one had a poor outcome which required below-knee amputation [Table 2].
| Discussion|| |
The exact incidence of Buerger's disease in India is not known, but it appears to be a common disease. It has a protracted and painful, but relatively benign clinical course. It usually occurs only in heavy smokers. It is characterized by the involvement of small- and medium-sized arteries. The digital arteries and the arteries of the calf and forearm may be occluded. The disease usually starts in one limb, but eventually both the lower limbs and upper limbs may be affected if the patients continue to smoke. In our study, all patients were heavy smokers of bidis or cigarettes for many years.
Various surgical modalities are used for the treatment of severe TAO. Arterial reconstruction could be done only in segmental proximal occlusions, but not in the distal arteries which are more commonly involved. In such areas, microvascular transplantation of free omental grafts and pedicled omental grafts has been successfully employed.
Amputation is indicated after gangrene formation, but it usually heals satisfactorily unlike that in arteriosclerosis.
Since distraction osteogenesis has been shown to stimulate neoangiogenesis, some studies have explored the use of this technique in the treatment of TAO;,,, it has, however, not been widely reported. We have used this technique in ten patients with acceptable results.
Corticotomy of the tibia with distraction causes neohistiogenesis and increase in collateral circulation in the limb. Ilizarov originally used the posteromedial cortex for distraction; the ASAMI group recommended distraction of the lateral cortex, and this was the method used in our study.
In the present series, good-to-excellent results were achieved in 8 (80%) cases, and these results were comparable with the findings of previous studies by Chaudhary et al. (90%), Patwa and Krishnan. (80%), Kulkarni et al. (83.3%), and Chouhan et al. (88%).,,,
Pin site infection was seen in 3 (30%) patients which were controlled by oral antibiotics and dressing. No deep infection was seen. Chouhan et al. observed pin tract infection in 16% of their cases.
No fractures during surgery or follow-up were seen in our study; however, in other such studies done with a larger number of cases, a small percentage had this complication (Patwa and Krishnan 3.33%; Chouhan et al. 8%); hence, its possibility should be kept in mind while operating.,
As in previous studies (Chouhan et al. 12%, Kulkarni et al. 13.33%, Patwa and Krishnan. 3.33%, and Chaudhary et al. 10%), in our study also, one of the patients (10%) showed no improvement and led to below-knee amputation.,,,
The limitations of this study include a very small number of patients, lack of long-term follow-up, and absence of control group.
| Conclusion|| |
In the absence of a well-accepted treatment of this limb-threatening and debilitating disease, neoangiogenesis by corticotomy and distraction offers a relatively simple and cost-effective surgical option to salvage ischemic limbs from amputations and provide pain relief, ischemic ulcer healing, and improved claudication distance, thus improving the quality of life. Histogenesis by horizontal distraction can be offered as a salvage procedure in patients with Buerger's disease, provided they are motivated for cessation of smoking.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Herrington JL Jr., Grossman LA. Surgical lesions of the small and large intestine resulting from Buerger's disease. Ann Surg 1968;168:1079-87.
Buerger L. Landmark publication from the American Journal of the Medical Sciences, 'Thrombo-angiitis obliterans: A study of the vascular lesions leading to presenile spontaneous gangrene'. 1908. Am J Med Sci 2009;337:274-84.
Wheeler HB. Thromboangitis obliterans. In: Sabiston DC Jr., editor. Textbook of Surgery, the Biological Basis of Modern Surgical Practice. 14th
ed. Philadelphia: WB Saunders Company; 1991. p. 1637-40.
Motukuru V, Suresh KR, Vivekanand V, Raj S, Girija KR. Therapeutic angiogenesis in Buerger's disease (thromboangiitis obliterans) patients with critical limb ischemia by autologous transplantation of bone marrow mononuclear cells. J Vasc Surg 2008;48:53S-60S.
Lie JT. Thromboangiitis obliterans (Buerger's disease) in women. Medicine (Baltimore) 1987;66:65-72.
Olin JW, Shih A. Thromboangiitis obliterans (Buerger's disease). Curr Opin Rheumatol 2006;18:18-24.
Mills JL Sr. Buerger's disease in the 21st
century: Diagnosis, clinical features, and therapy. Semin Vasc Surg 2003;16:179-89.
Chalon S, Moreno H Jr., Benowitz NL, Hoffman BB, Blaschke TF. Nicotine impairs endothelium-dependent dilatation in human veins in vivo
. Clin Pharmacol Ther 2000;67:391-7.
Bozkurt AK, Köksal C, Demirbas MY, Erdoğan A, Rahman A, Demirkiliç U, et al.
A randomized trial of intravenous iloprost (a stable prostacyclin analogue) versus lumbar sympathectomy in the management of Buerger's disease. Int Angiol 2006;25:162-8.
Cotton RC, Craven JL. Treatment of thromboangiitis obliterans with clofibrate (Atromid-S). Angiology 1968;19:307-10.
Saha K, Chabra N, Gulati SM. Treatment of patients with thromboangiitis obliterans with cyclophosphamide. Angiology 2001;52:399-407.
Piazza G, Creager MA. Thromboangiitis obliterans. Circulation 2010;121:1858-61.
Sasajima T, Kubo Y, Inaba M, Goh K, Azuma N. Role of infrainguinal bypass in Buerger's disease: An eighteen-year experience. Eur J Vasc Endovasc Surg 1997;13:186-92.
Sayin A, Bozkurt AK, Tüzün H, Vural FS, Erdog G, Ozer M. Surgical treatment of Buerger's disease: Experience with 216 patients. Cardiovasc Surg 1993;1:377-80.
Shionoya S. Buerger's disease: Diagnosis and management. Cardiovasc Surg 1993;1:207-14.
Nishimura A, Sano F, Nakanishi Y, Koshino I, Kasai Y. Omental transplantation for relief of limb ischemia. Surg Forum 1977;28:213-5.
Donas KP, Schulte S, Ktenidis K, Horsch S. The role of epidural spinal cord stimulation in the treatment of Buerger's disease. J Vasc Surg 2005;41:830-6.
Isner JM, Baumgartner I, Rauh G, Schainfeld R, Blair R, Manor O, et al.
Treatment of thromboangiitis obliterans (Buerger's disease) by intramuscular gene transfer of vascular endothelial growth factor: Preliminary clinical results. J Vasc Surg 1998;28:964-73.
Saito S, Nishikawa K, Obata H, Goto F. Autologous bone marrow transplantation and hyperbaric oxygen therapy for patients with thromboangiitis obliterans. Angiology 2007;58:429-34.
Matoba S, Tatsumi T, Murohara T, Imaizumi T, Katsuda Y, Ito M, et al.
Long-term clinical outcome after intramuscular implantation of bone marrow mononuclear cells (therapeutic angiogenesis by cell transplantation [TACT] trial) in patients with chronic limb ischemia. Am Heart J 2008;156:1010-8.
Tavakoli H, Salimi J, Rashidi A. Reply: “Treatment-of-choice for Buerger's disease (thromboangiitis obliterans): Still an unresolved issue”. Clin Rheumatol 2008;27:813.
Ilizarov GA. The tension-stress effect on the genesis and growth of tissues. Part I. The influence of stability of fixation and soft-tissue preservation. Clin Orthop Relat Res 1989;(238):249-81.
Ohashi S, Ohnishi I, Kageyama T, Imai K, Nakamura K. Distraction osteogenesis promotes angiogenesis in the surrounding muscles. Clin Orthop Relat Res 2007;454:223-9.
Ilizarov GA. The tension-stress effect on the genesis and growth of tissues: Part II. The influence of the rate and frequency of distraction. Clin Orthop Relat Res 1989;(239):263-85.
Patwa JJ, Krishnan A. Buerger's disease (thromboangiitis obliterans)- management by ilizarov's technique of horizontal distraction. A retrospective study of 60 cases. Indian J Surg 2011;73:40-7.
Chaudhary M, Chaudhary P, Chaudhary MR. Treatment of TAO, by bone widening using Ilizarov technique. Indian J Surg 2001;63:389-92.
Kulkarni S, Kulkarni G, Shyam AK, Kulkarni M, Kulkarni R, Kulkarni V. Management of thromboangiitis obliterans using distraction osteogenesis: A retrospective study. Indian J Orthop 2011;45:459-64.
] [Full text]
Chouhan A, Meena DS, Meena UK, Behera P, Yadav L, Gupta V. Limb salvage in Buerger's disease by distraction histogenesis: A prospective study with literature review. J Clin Orthop Trauma 2019;10:981-5.
Shionoya S, Hirai M, Kawai S, Seko T, Ban I. Pattern of arterial occlusion in Buerger's disease. Angiology 1982;33:375-84.
Association for the Study and Application of Ilizarov's Method (A.S.A.M.I. Group). Bianchi-Maiocchi A, Aronson J, editors. Widening and reconstruction of leg. In: Operative Principles of Ilizarov. Baltimore; Williams and Wilkins: 1991. p. 456.
[Figure 1], [Figure 2], [Figure 3], [Figure 4]
[Table 1], [Table 2]