Compartment syndrome of the non-injured limb

Abstract

Compartment syndrome is a common limb-threatening entity in trauma. However, the occurrence of the same in the non-injured limb is rare. It seems to be multifactorial in origin, with abnormal positioning being the most common cause. We present such a case of well-leg compartment syndrome which was treated by an urgent fasciotomy. We emphasise on the fact that the diagnosis of compartment syndrome is clinical and the management remains the same irrespective of whether the limb has sustained an injury or not.

Background

Compartment syndrome of the limb is a well-known entity. Several causes of compartment syndrome have been described in the literature with trauma to the extremity being the most common. Well-leg compartment syndrome (WLCS) is a subset of compartment syndrome which was first described by Heppenstall et al in 1999.¹ It is caused by abnormal positioning during surgery, in contrast to a classical compartment syndrome caused by trauma.² Other contributing factors for the development of WLCS have recently been described.² However, this condition remains underdiagnosed and under-reported. We present a case of WLCS of the non-injured limb in a young boy following trauma and its successful management.

Case presentation

An 18-year-old boy was brought to the emergency department (ED) 3 hours following a road traffic injury due to two-wheeler skid. He was a pillion rider and was non-helmeted. The bike fell over his left lower limb, but there was no history of prolonged extrication. He was triaged to the yellow area of the resuscitation bay based on a low velocity of the mechanism of injury and haemodynamically stable status. On the initial assessment, the airway was patent, the breathing was spontaneous, the pulse rate was 84 per min and blood pressure was 110/70 mm Hg. His Glasglow Coma Scale was E4V5M6 and he was moving all four limbs. X-rays of the chest and pelvis were within normal limits. Focused Assessment by Sonography in Trauma examination was negative. He was fully alert and complained of excruciating pain over his left thigh, especially on unintended movements during transport. The secondary survey revealed that he had a deformed left thigh with bony crepitus, without any overlying skin or soft tissue injury. The right lower limb showed no signs of bony or soft tissue injury, with no alteration in its sensory or motor functions. A Thomas knee splint was applied on the left side. X-ray of the left thigh showed a fractured shaft of the femur.

He underwent open reduction and internal fixation of left femur under combined spinal and epidural anaesthesia in a lithotomy position. In the postoperative period, after about 5 hours, the patient complained of painful swelling in his right leg (non-injured limb). There were no signs of any external injury. No intravenous cannula was applied in the right lower limb. He had a tense swelling in the calf region of his right leg. He was experiencing excruciating pain with a score of 8/10 on the Visual Analogue Scale (VAS) despite epidural and intravenous analgesia. There was motor and sensory dysfunction to his right foot, which made him unable to move the toes. However, distal pulses in both the lower limbs were palpable and equal.

Investigations

A clinical diagnosis of compartment syndrome of the right leg with unknown aetiology was made.

Differential diagnosis

Deep venous thrombosis was ruled out using ultrasound and colour Doppler examination (figure 1).

Figure 1.

Colour Doppler study showing normal blood flow in deep veins of non-injured right lower limb.

Treatment

The patient underwent a two-incision, four-compartment fasciotomy of the right leg (figure 2). Edematous and tense muscles with bulge were noted intraoperatively (figure 3).

Figure 2.

Compartment syndrome of the right leg planned for two-incision, four-compartment fasciotomy: (A) medial incision marking and (B) marking of fibular line for lateral incision.

Figure 3.

Intraoperative picture showing muscles bulging through the incision after fascia is incised.

Outcome and follow-up

Patient dramatically improved clinically in the immediate postoperative period. His pain significantly reduced with the VAS score of 2/10. There was an improvement of the motor functions of his right foot with the return of toe movements within 12 hours. His sensory deficit also gradually improved in a week. Fasciotomy wounds were closed with split-thickness skin grafting. He was discharged on 15th postinjury day in good health.

Discussion

Compartment syndrome is characterised by an increase of pressure within a closed anatomical space. This increase in pressure results in a decrease in perfusion pressure and resultant ischemia which if left untreated may lead to limb loss and fatal systemic complications. This is most commonly seen in the legs, due to a high incidence of tibial fracture and the presence of four closed fascial spaces (compartments) in a small area. The four compartments in the leg are anterior, lateral, deep posterior and superficial posterior (table 1).^{3 4}

Table 1.

Anatomical description of compartments of the leg

Compartment	Muscles	Main vascular contents	Main neural contents
Anterior	Tibialis anterior, extensor digitorum longus, peroneus tertius and extensor hallucis longus	Anterior tibial artery	Deep peroneal nerve
Lateral	Peroneus longus and peroneus brevis	Branches from peroneal artery	Superficial peroneal nerve
Superficial posterior	Gastrocnemius, plantaris and soleus		Sural nerve
Deep posterior	Popliteus, flexor digitorum longus, flexor hallucis longus and tibialis posterior	Posterior tibial artery and peroneal artery	Tibial nerve

Pathophysiology of compartment syndrome was initially thought to be due to the direct plasma leak after ischemia resulting in the increased compartment pressure. The current concepts focus on the ischemia-reperfusion theory of cellular injury.⁵ Ischemia leads to depletion of intracellular energy stores, causing cellular oedema, increasing venular pressure and venous outflow obstruction.⁵ Eventually, it leads to a rise in intra-compartmental pressures and decreased perfusion to tissues.

Factors associated with the increased risk of compartment syndrome in the normal limb are enumerated below (table 2).

Table 2.

Factors associated with the increased risk of WLCS

Factor	Comments
Procedure duration	Surgeries longer than 6 hours pose a risk10
Patient position	Lloyd-Davies position elevates the limbs and angulates the hips, thereby causing a decrease in arterial perfusion pressure11 The more the body mass index, the more the decrease in ankle arterial pressure is11 Ankle dorsiflexion increases compartmental pressures12
Leg holders/ankle supports	Allen stirrups supporting the ankle may decrease compartment pressures, as opposed to calf supporting leg holders12
Stockings and IPCD	They increase compartment pressures and cause local hypoperfusion13
Head-down tilt	15° of head-down tilt in lithotomy causes ischaemia, followed by a postoperative period surge in reperfusion, causing the risk of WLCS14
Physiological insults during surgery	Hypovolemia (blood loss, insensible loss, and inadequate replacement) Hypotension (blood loss and acidemia) Vasoconstrictor drugs Sympathetic blockade
Epidural anaesthesia	Local anaesthetics increase blood flow after a sympathetic blockade, contributing to an increase in compartmental pressures15 Masks early symptom of pain in compartment syndrome16
Peripheral vascular disease	A proper history of smoking, intermittent claudication to be taken Clinical examination and arterial Doppler study should be done

IPCD, intermittent pneumatic compression devices; WLCS, well-leg compartment syndrome.

In our case, the duration of surgery was 2.5 hours, the head end of the operating table was not tilted during surgery, no stockings or intermittent pneumatic compression devices were used and there was no physiological insult during surgery. However, the patient was operated in the lithotomy position, and leg holders were used. Postoperatively epidural analgesia was used. Thus, compartment syndrome in our case seems to be multifactorial in which lithotomy position caused the initial ischaemia followed by reperfusion after surgery. The epidurally administered bupivacaine added on to it by increasing the postoperative blood flow even more.

There is a dearth of literature on WLCS because many cases go unreported due to misdiagnosis or subclinical symptoms.² We did a thorough online Medline search. We searched with the headings of WLCS, compartment syndrome in non-injured limb and compartment syndrome due to operative intervention. We further excluded the cases in which compartment syndrome was found in an injured limb. We found that only 41 cases have been reported so far in English literature over the last 50 years (table 3). Recently, the number of reported cases has increased (figure 4). This suggests that this pathology may be very much under-reported and clinicians should be aware of this pathology.

Table 3.

Reported cases of WLCS in literature in the last few decades

	Author	Year	Surgery	Position	Duration (hours)	Physiological insults	Leg	Management	Time of diagnosis	Morbidity
1	Reddy et al16	1984	Urological surgery	Metal skid	Not known	Nil	B/L	Fasciotomy	Several hours	Motor dysfunction
2	Werbel et al17	1986	Urological surgery	Lithotomy with IPCD	9	Malfuction of IPCD	L	Fasciotomy	Not known	Sensory dysfunction
3	Dugdale et al (two cases)18	1989	Orthopaedic surgery	Supine	5.75 and 6	Nil	R	Fasciotomy	Not known	Sensory and motor deficits
4	Bergqvist et al19	1990	Urological surgery	Not known	8	Not known	B/L	Fasciotomy	21 hours	Sensory and motor dysfunction
5	Montgomery et al (two cases)20	1991	Urological surgery	Lithotomy	10.5 and 11.5	Epidural analgesia	R, B/L	Fasciotomy	18.5 hours and 16 hours	Sensory and motor dysfunction
6	Crinnion et al21	1996	Urological surgery	Lloyd-Davies	4.5	Not known	B/L	Not known	Not known	Not known
7	Beerle et al22	1993	Urological surgery	High lithotomy	7.5	Not known	Not known	Not known	Not known	Not known
8	Anglen et al (two cases)23	1994	Orthopaedic surgery	Lithotomy	6 and 6.25	Not known	Not known	Fasciotomy	>24 hours	Sensory and motor dysfunction
9	Moses et al24	1994	Urological surgery	High lithotomy	5	Epidural analgesia	B/L	Fasciotomy	>24 hours	Sensory and motor dysfunction
10	Carlson et al (two cases)25	1995	Orthopaedic surgery	Hemilithotomy	3.5	Not known	B/L	Not specified	16 hours and 20 hours	Motor dysfunction
11	Tuncer et al26	1997	Urological surgery	Lloyd-Davies	7	Not known	B/L	Fasciotomy	15 hours	Complete recovery
12	Adler et al (three cases)27	1997	Orthopaedic surgery	Hemilithotomy	5.5, 7 and 4.5	Not known	L, R	Fasciotomy	1 hour	Sensory dysfunction
13	Ikeya et al28	2006	Colorectal surgery	Lithotomy	7.35	Nil	B/L	Fasciotomy	Within 24 hours	Complete recovery
14	Turnbull et al (three cases)29	2008	Urological surgery	Lloyd-Davies	7.5	Epidural analgesia	B/L	Fasciotomy	Recovery area	Sensory and motor dysfunction
15	Krarup et al30	2008	Colorectal surgery	Lithotomy	4.83	Not known	L	Fasciotomy	Within 24 hours	Sensory dysfunction
16	Weber et al31	2008	Orthopaedic surgery	Hemilithotomy	2.92	Not known	L	Fasciotomy	18 hours later	Sensory and motor dysfunction
17	Noordin et al32	2009	Orthopaedic	Lloyd-Davies Leg in well-padded calf rest	4.25	Nil	R	Fasciotomy	14 hours postop	Complete recovery
18	Pollard et al33	2009	Breast surgery	Supine	9	Nil	L	No fasciotomy	Recovery area	Pain and leg cramps
19	Raman et al34	2009	Robotic prostatectomy	Lithotomy trendelenburg	5	Nil	B/L	Fasciotomy	Immediate postop	Sensory dysfunction
20	Chin et al35	2009	Gastrointestinal surgery	Lloyd-Davies	7	Nil	B/L	Fasciotomy	2 days	Sensory and motor dysfunction
21	Teeples et al36	2010	Maxillomandibular surgery	Supine	5.5	Hypotension and drugs	L	Fasciotomy	>24 hours	Complete recovery
22	O’Connor et al37	2010	Upper limb vascular surgery	Supine DVT stockings	15	Not known	L	Not known	Not known	Not known
23	Takano et al30	2012	Colorectal surgery	Lithotomy	6.82	Not known	L	Fasciotomy	Within 24 hours	Sensory dysfunction
24	Tashakkor et al38	2012	Breast surgery	Supine	12.5	Nil	R	Fasciotomy	48 hours	Motor dysfunction
25	Malahias et al39	2012	Breast Surgery	Supine	4.25	Nil	B/L	Fasciotomy	1 hour	Not known
26	Boesgaard-kjer et al40	2013	Gynaecological surgery	Lithotomy	5	Nil	B/L	Fasciotomy	5 hours	Sensory dysfunction
27	Boesgaard-kjer et al40	2013	Gynaecological surgery	Lithotomy	5	Nil	R	No fasciotomy	7 hours	Motor dysfunction
28	Yoshimura et al30	2014	Colorectal surgery	Lithotomy	12.12	Not known	L	Fasciotomy	Within 24 hours	Sensory dysfunction
29	Munakata et al3	2014	Colorectal surgery	Lithotomy	5.5	Not known	L	Fasciotomy	Within 24 hours	Complete recovery
30	Uji et al (two cases)30	2014	Colorectal surgery	Lithotomy	8.75 and 7.58	Not known	L in both	No fasciotomy	Within 12 hours	Complete recovery
31	Meena et al41	2014	Orthopaedic surgery	Lithotomy Leg in leg holder	2.5	Permissive hypotension	R	Fasciotomy	4 hours postop	Complete recovery
32	Ozawa et al30	2015	Colorectal surgery	Lithotomy	6.42	Not known	L	Fasciotomy	Immediately	Complete recovery
33	Enomoto et al42	2016	Colorectal surgery	Lithotomy	8.15	Nil	B/L	Fasciotomy	24 hours	Sensory dysfunction
34	Konishi et al30	2016	Colorectal surgery	Lithotomy	5.47	Not known	L	Fasciotomy	Immediately	Complete recovery
35	Uetaki et al30	2016	Colorectal surgery	Lithotomy	12.35	Not known	L	Fasciotomy	Immediately	Complete recovery
36	Stornelli et al43	2016	Gynaecological surgery	Lithotomy trendelenburg	1.5	Rhabdomyolysis	B/L	Fasciotomy	Immediately	Complete recovery
37	Clarke et al44	2017	Orthopaedic surgery	Hemilithotomy	6.25	Rhabdomyolysis	L	Fasciotomy	Immediately	Complete recovery
38	Nishino et al30	2017	Colorectal surgery	Lithotomy	11.4	Nil	L	Fasciotomy	15 hours	Complete recovery
39	Yamamoto et al45	2018	Robotic cystectomy	Lithotomy	6.98	Nil	B/L	Fasciotomy	20 hours	Complete recovery
40	Brouze et al46	2019	Orthopaedic surgery	Hemilithotomy	6	Hypotension	R	Fasciotomy	2 hours	Complete recovery
41	Brouze et al46	2019	Orthopaedic surgery	Hemilithotomy	5.65	Multiple hypotensive episodes	L	Fasciotomy	1 hour	Complete recovery

DVT, deep vein thrombosis; IPCD, intermittent pneumatic compression devices; WLCS, well-leg compartment syndrome.

Figure 4.

Bar diagram showing the distribution of cases over the last 50 years. WLCS, well-leg compartment syndrome.

Ours is a high-volume level 1 trauma centre with an annual footfall of more than 70 000 patients in ED. We manage a good number of patients with vascular injuries with or without compartment syndrome. A high index of clinical suspicion is the key to the management of compartment syndrome. Pain out of proportion, tense swelling in the calf region and paresthesia are good enough signs to diagnose extremity compartment syndrome. A palpable distal pulse should not exclude compartment syndrome. Furthermore, missed compartment syndrome has high morbidity in the form of limb loss. Irrespective of the cause, the diagnosis and management of compartment syndrome remain the same. Early fasciotomy has been shown to improve the outcome. Debakey et al described the two-incision, four-compartment fasciotomy for the leg.⁶ It involves two incisions: (a) anterolateral incision: a longitudinal incision is made midway between tibia and fibula to open up anterior and lateral compartments and (b) posteromedial incision: a longitudinal incision is made 2 cm posterior to the medial border of the tibia to expose superficial posterior compartment. The deep posterior compartment is exposed by detaching soleal attachment from the tibia. Ngheim and Boland advocated a single incision, four-compartment fasciotomy.⁷ They used a single anteromedial incision to approach all the four compartments. They approached the superficial posterior compartment through the intermuscular septum along the posterior wall of the lateral compartment, while the deep posterior compartment was approached by incising the intraosseous membrane. Several other variations of the techniques have been described, but the superiority of one method over others has not been established.⁸ Having said that, it is important to note that, whichever method is chosen, the opening of all the four compartments is necessary for acute compartment syndrome of the leg.

As with every other surgical procedure, fasciotomy is also associated with complications in the shape of soft tissue infections, paraesthesia, chronic pain, contractures, ulcerations and osteomyelitis. Fitzgerald et al reported that 81% of patients undergoing fasciotomies develop long-term complications.⁹ As such, early closure of fasciotomy is now recommended, which has been supplemented by the use of negative pressure wound therapy. However, the authors want to emphasise that complications of fasciotomy should not deter a surgeon when the clinical condition of patient demands it. At the same time, it should be judiciously used based on the sound clinical examination. Our case highlights the importance of early detection of compartment syndrome and its management even in a non-injured limb. It describes a rare entity of WLCS and brings into light the importance of the thorough physical examination and the tertiary survey in trauma patients.

Patient’s perspective.

I was admitted with a left leg fracture and underwent surgery for the same. But, suddenly I started having severe pain in my normal leg. Initially, I thought that it must be some missed injury, but then I could not even straighten my ankle. I was taken for another surgery immediately, after which the pain was gone and my leg started working fine. I thank my doctors for prompt treatment.

Learning points.

• Cause of well-leg compartment syndrome is multifactorial, and identification of risk factors is essential.

• Compartment syndrome is a clinical diagnosis, and a keen clinical eye helps in picking up the subtle signs.

• Management of compartment syndrome remains the same irrespective of the cause.

• Early fasciotomy is indicated for any type of compartment syndrome, be it in the injured leg or the normal leg.