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1068 CASE REPORTS

THE JOURNAL OF BONE AND JOINT SURGERY

tive accuracy. External fixation 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 benefits 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.

References1. 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 tibialdeformities. Tech Orth 1997;12:260-75.

4. Paley D, Herzenberg JE, Tetsworth K, McKie J, Bhave A. Deformity planning forfrontal 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 Ili za-

roc technique. Clin Orthop 1994;301:38-41.

■ CASE REPORT

A spontaneous compartment syndrome in apatient 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 Officer

N. Viswanathan, MB BS,

FRCS, Registrar

E. Aldlyami, MBChB,MRCS, Senior House Officer

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 andOrthopaedics, 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 defined 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 diabetesmellitus,1-4 hypothyroidism,5 influenza-virus-induced

myositis,6 leukaemic infiltration,7 the nephrotic syn-

drome,8 a ruptured aneurysm,9 anticoagulation10

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 fluid pressure

in the osteofascial compartment, vascular occlusion

and muscle necrosis.

Case reportA 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 definite

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 dorsiflex hisfoot. 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 109 /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 inflammation. There were some viable

atrophic muscle fibres (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.

DiscussionSpontaneous compartment syndrome has been reported in influen-

zal myositis, hypothyroidism, leukaemic infiltration, 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 al1 described a similar case when the

patient was operated on within eight hours of the onset of symp-

toms. He suffered no neurological deficit. Smith and Laing2

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

deficit 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 conflicting views regarding the development of

spontaneous compartment syndrome in diabetics. One suggests

that metabolic disturbances cause osmotic accumulation of fluid

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 alocalised 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 viablemuscle fibres possessing nuclei (haematoxylin and eosin, x2).

Fig. 2

A medium calibre septal blood vessel showing recanalisation withfocal, 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 benefits 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.

References1. 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 lowerextremity 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 hypothyroidpatient. Thyroid 1995;5:305-8.

6. Paletta CE, Lynch R, Knutsen AP. Rhabdomyolysis and lower extremity compart-ment syndrome due to influenza B virus. Ann Plast Surg 1993;30:272-3.

7. Veeragandham RS, Paz IB, Nadeemanee A. Compartment syndrome of the legsecondary to leukemic infiltration: a case report and review of literature. J Surg Oncol 1994;55:198-200.

8. Sweeney HE, O’Brien 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 byruptured tibial artery aneurysm in a patient with polyarteris nodosa: a case report andreview of literature. J Vasc Surg 1993;18:295-8.

10. Griffiths 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 tibiofibular 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 tibialcompartment 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 diabetesmellitus. 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.

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