1068 CASE REPORTS

THE JOURNAL OF BONE AND JOINT SURGERY

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.
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patient. Thyroid 1995;5:305-8.
6. Paletta CE, Lynch R, Knutsen AP. Rhabdomyolysis and lower extremity compart-
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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.
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ruptured tibial artery aneurysm in a patient with polyarteris nodosa: a case report and
review of literature. J Vasc Surg 1993;18:295-8.
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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.