tive accuracy. External xation allows per- and post-operative adjustment and thus high accuracy, but poor comfort. FAN has a steep learning curve. Good quality intra-operative radiographs are a prerequisite for accuracy. The total treatment time is less than with other techniques. In these two cases, the tech- nique did not create any limb length inequality. No benets in any form have been received or will be received from a commer- cial party related directly or indirectly to the subject of this article. References 1. Smyth EHJ. Windswept deformity. J Bone Joint Surg [Br] 1980;62-B:166-7. 2. Mankin HJ. Rickets, osteomalacia and renal osteodystrophy: an update. Orthop Clin North Am 1990;21:81-96. 3. Paley D, Herzenberg JE, Bor N. Fixator assisted nailing of femoral and tibial deformities. Tech Orth 1997;12:260-75. 4. Paley D, Herzenberg JE, Tetsworth K, McKie J, Bhave A. Deformity planning for frontal and sagittal plane corrective osteotomies. Orthop Clin North Am 1994;25:425-65. 5. Stanitski DF. Treatment of deformity secondary to metabolic bone disease with Iliza- roc technique. Clin Orthop 1994;301:38-41. CASE REPORT A spontaneous compartment syndrome in a patient with diabetes R. M. Jose, N. Viswanathan, E. Aldlyami, Y. Wilson, N. Moiemen, R. Thomas From Department of Plastic Surgery, Selly Oak Hospital, Birmingham, UK R. M. Jose, MB BS, MCh, FRCS, Senior House Ofcer N. Viswanathan, MB BS, FRCS, Registrar E. Aldlyami, MBChB, MRCS, Senior House Ofcer Y. Wilson, MBChB, FRCS, Consultant N. Moiemen, MBBCh, FRCS, Consultant Department of Plastic Surgery, Selly Oak Hospital, Birmingham B29 6JD, UK. R. Thomas, MB BS, MRCS, LRCP, Consultant Department of Trauma and Orthopaedics, New Cross Hospital, Wolverhampton WV10 0QP, West Midlands, UK. Correspondence should be sent to Mr R. M. Jose. 2004 British Editorial Society of Bone and Joint Surgery doi:10.1302/0301-620X.86B7. 14770 $2.00 J Bone Joint Surg [Br] 2004;86-B:1068-70. Received 9 July 2003; Accepted after revision 16 October 2003 A compartment syndrome is an orthopaedic emergency which can result from a variety of causes, the most common being trauma. Rarely, it can develop spontaneously and several aetiologies for spontaneous compartment syndrome have been described. We describe a patient with diabetes who developed a spontaneous compartment syndrome. The diagnosis was delayed because of the atypical presentation. Compartment syndrome is dened as an elevation of the interstitial pressure in a closed osteofascial com- partment causing microvascular compromise. The common causes include trauma, arterial injury, limb compression and burns. Rarely, it can also occur spontaneously in association with type-I diabetes mellitus, 1-4 hypothyroidism, 5 inuenza-virus-induced myositis, 6 leukaemic inltration, 7 the nephrotic syn- drome, 8 a ruptured aneurysm, 9 anticoagulation 10 and a ganglion cyst. 11 Four cases of spontaneous compartment syndrome in diabetics have been described previously and many theories regarding the aetiology have been advanced, including meta- bolic changes giving rise to increased uid pressure in the osteofascial compartment, vascular occlusion and muscle necrosis. Case report A 47-year-old man of Asian origin developed pain in the anterolateral aspect of the left leg after a brief walk. It was moderate in intensity but was not relieved by rest. He had suffered from type-I diabe- tes mellitus, well controlled on insulin, for almost 20 years. He was also hypertensive and was undergoing laser treatment for diabetic retinopathy. He attended the Emergency Department with a localised red, tender area over the upper lateral aspect of the left leg below the knee. No denite diagnosis was made and he was given analgesics and discharged. The pain was not relieved and he was prescribed stronger analgesics by his general practi- tioner. The pain increased in intensity over the next four days and he developed foot drop. He was seen again and referred for an orthopaedic opinion. There was swelling, redness and tenderness over the anterolateral aspect of the left leg. He had normal sensation but was unable to dorsiex his foot. Both the dorsalis pedis and posterior tibial pulses were present. The differential diagnoses were an intrafascial bleed, infection, spontaneous muscle necrosis or a compartment syndrome. Haematological investigation revealed a mild leu- kocytosis (12.8 x 10 9 /l). Biochemical analysis was normal except that the level of creatine kinase was increased to 4178 U/l, raising the suspicion of muscle necrosis and a compartment syndrome. Decompression of the anterior and lateral compart- ments was carried out. The muscles were found to bulge beneath the deep fascia and the compartmen- tal pressure was raised. Both muscle groups appeared to be ischaemic and did not respond to pinching. The pain persisted and he was taken back to theatre after two days. Necrotic parts of tibialis anterior were excised and sent for histological examination. The wound was left open and dressed regularly. At one week it was closed secondarily, without a skin graft. Histological examination of the excised specimen showed areas of devitalised skeletal muscle without evidence of inammation. There were some viable atrophic muscle bres (Fig. 1) with blood vessels showing thrombus and recanalisation (Fig. 2). CASE REPORTS 1069 VOL. 86-B, No. 7, SEPTEMBER 2004 He was reviewed in the Outpatient Clinic after two weeks when his wound had healed. There has been no improvement in the foot drop. He continues to attend for physiotherapy and a tendon transfer is being considered. Discussion Spontaneous compartment syndrome has been reported in inuen- zal myositis, hypothyroidism, leukaemic inltration, nephrotic syndrome, vascular anomalies, anticoagulant therapy and cystic lesions. 5-11 There have been four other case reports of spontaneous compartment syndrome in diabetes mellites. 1-4 In 1997 Chautems et al 1 described a similar case when the patient was operated on within eight hours of the onset of symp- toms. He suffered no neurological decit. Smith and Laing 2 reported a case of bilateral compartment syndrome in a diabetic patient who presented to the Emergency Department after four days. He was found to have muscle necrosis, a bilateral sensory decit in the distribution of the deep peroneal nerve, and a foot drop. The delay in the diagnosis of compartment syndrome in our patient may be excused by its atypical presentation. Initially, he had localised swelling and only moderate pain. Absence of pain has been reported previously by Ciacci et al, 12 who suggested a possible neurapraxic block of the deep peroneal nerve as an expla- nation. There are two conicting views regarding the development of spontaneous compartment syndrome in diabetics. One suggests that metabolic disturbances cause osmotic accumulation of uid in the muscle which may be the primary event leading to increased pressure. 13 The muscle necrosis develops as a result of the ischaemia. 14 The other view is that spontaneous muscle in- farction, because of microvascular blockage, is the primary event and that compartmental pressures rise subsequent to that. 2,4 We prefer the latter explanation since our patient had a localised swelling initially and the symptoms progressed over several days. The histopathology of the excised muscle showed thrombi in the small blood vessels with attempts at recanalisa- tion (Fig. 2). A relevant coincidence is that our patient, and two other reported patients, had diabetic retinopathy which suggests coexisting microvascular disease. There have been other record- ed cases of spontaneous muscle infarction in diabetics. They are common in type-I diabetes and are strongly associated with other microvascular complications such as neuropathy, retino- pathy and nephropathy. 15 The usual presentation has been a swelling in the muscles of the thigh and the treatment has Fig. 1 Necrotic pale muscle bundles bereft of nuclei surrounded by viable muscle bres possessing nuclei (haematoxylin and eosin, x2). Fig. 2 A medium calibre septal blood vessel showing recanalisation with focal, residual intraluminal thrombus (haematoxylin and eosin, x10) 1070 CASE REPORTS THE JOURNAL OF BONE AND JOINT SURGERY mostly been conservative. 16,17 Since the compartment in the calf is smaller and tighter, swelling within it can easily result in a compartment syndrome. Early surgery is more likely to be cur- ative. No benets in any form have been received or will be received from a commer- cial party related directly or indirectly to the subject of this article. References 1. Chautems RC, Irmay F, Magnin M, Morel P, Hoffmeyer P. Spontaneous anterior and lateral tibial compartment syndrome in type 1 diabetic patient: case report. J Trauma 1997;43:140-1. 2. Smith AL, Laing PW. Spontaneous compartment syndrome in Type 1 diabetes mel- litus. Diabet Med 1999;16:168-9. 3. Lecky B. Acute bilateral anterior tibial compartment syndrome after caesarian sec- tion in a diabetic. J Neurol Neurosurg Psychiatry 1980;43:88-90. 4. Parmoukian VN, Rubino F, Iraci JC. Review and case report of idiopathic lower extremity compartment syndrome and its treatment in diabetic patients. Diabetes Metab 2000;26:489-92. 5. Hsu SI, Thadhani RI, Daniels GH. Acute compartment syndrome in a hypothyroid patient. Thyroid 1995;5:305-8. 6. Paletta CE, Lynch R, Knutsen AP. Rhabdomyolysis and lower extremity compart- ment syndrome due to inuenza B virus. Ann Plast Surg 1993;30:272-3. 7. Veeragandham RS, Paz IB, Nadeemanee A. Compartment syndrome of the leg secondary to leukemic inltration: a case report and review of literature. J Surg Oncol 1994;55:198-200. 8. Sweeney HE, OBrien F. Bilateral anterior tibial compartment syndrome in associa- tion with nephrotic syndrome: report of a case. Arch Intern Med 1965;116:487-90. 9. Hasaniya N, Katzen JT. Acute compartment syndrome of both lower legs caused by ruptured tibial artery aneurysm in a patient with polyarteris nodosa: a case report and review of literature. J Vasc Surg 1993;18:295-8. 10. Grifths D, Jones DH. Spontaneous compartment syndrome in a patient on long- term anticoagulation. J Hand Surg [Br] 1993;18:41-2. 11. Ward WG, Eckardt JJ. Ganglion cyst of the proximal tibiobular joint causing ante- rior compartment syndrome. J Bone Joint Surg [Am] 1994;76-A:1561-4. 12. Ciacci G, Federico A, Giannini F, et al. Exercise-induced bilateral anterior tibial compartment syndrome without pain. Ital J Neurol Sci 1986;7:377-80. 13. Coley S, Situnayaki RD, Allen MJ. Compartment syndrome, stiff joints, and dia- betic cheiroarthropathy. Ann Rheum Dis 1993;52:840. 14. Chester CS, Banker BWQ. Focal infarction of muscle in diabetics. Diabetic Care 1986;9:623-30. 15. Grigoriadis E, Fam AG, Starok M, Ang LC. Skeletal muscle infarction in diabetes mellitus. J Rheum 2000;27:1063-8. 16. Lauro GR, Kissel JT, Simon SR. Idiopathic muscular infarction in a diabetic patient. J Bone Joint Surg [Am] 1991;73-A:301-4. 17. Banker BQ, Chester CS. Infarction of the thigh muscle in the diabetic patient. Neu- rology 1973;23:667-77.