Compartment syndrome of the thigh is an infrequent phenomenon, and it is
even more rare for it to occur as a result of exercise. The vast majority of
cases are associated with femoral fractures, thigh contusions, prolonged
compression, and/or vascular
injuries1,2.
A search of the English-language literature revealed only eleven cases caused
by intense or prolonged exercise (Table
I). More importantly, we found only one reported case of bilateral
thigh and leg compartment syndrome, the outcome of which was death secondary
to multiorgan
failure3. We are
presenting what we believe is the first report on a survivor of
exercise-induced compartment syndrome of both thighs and legs. Follow-up at
fourteen months demonstrated reasonable functional recovery despite
life-threatening medical complications and the development of massive
heterotopic ossification.
The patient agreed to have the data concerning the case submitted for
publication.
A twenty-two-year-old male body-builder had been taking creatine
monohydrate (20 g/day) as a nutritional supplement for four months when acute
thigh and leg cramping developed, followed by five days of the passage of dark
urine. The symptoms developed over a two-week period while the man was
performing high-repetition, free-weight lower-extremity exercises. He was
admitted to a local community hospital with symptoms of nausea, vomiting,
uncontrollable thigh pain, and the inability to walk. Three days after
admission, he was transferred to our institution with a delayed diagnosis of
acute compartment syndrome of the thighs. Prior to his transfer, anterior
thigh compartment pressure was reported to be elevated bilaterally (65 mm
Hg).
Physical examination performed following the transfer demonstrated tense
thighs and legs. The clinical symptoms, however, appeared to have abated
slightly compared with the initial presentation three days previously. The
patient did not have pain with passive muscle stretch and his use of narcotics
had diminished considerably since his transfer from the referring hospital.
The distal pulses were strong and symmetric, and the neurological examination
revealed normal findings. Compartment pressures were not measured.
Table II summarizes the
laboratory data.
The presentation was consistent with a delayed diagnosis of acute
compartment syndrome of the thighs, myonecrosis, and myoglobinuria. After a
thorough review of the clinical course, we chose to follow the patient
expectantly, in accordance with current recommendations regarding management
of a delayed presentation of acute compartment syndrome, as the performance of
fasciotomy at this stage was thought to be associated with a prohibitive risk
of infection due to exposure of necrotic muscle to the
environment4. Given
the extent of the myoglobinuria and the concern about rhabdomyolysis, the
patient was managed by the medicine service and was treated with aggressive
hydration. His clinical condition worsened dramatically over a
twenty-four-hour period, with an increase in bilateral thigh swelling, pain
with passive quadriceps stretch, increased narcotic use, and worsening
myonecrosis (Table II).
Compartment pressures were not measured.
The patient underwent bilateral fasciotomy of the anterior (quadriceps) and
posterior (hamstrings) compartments of the thigh through a lateral incision on
the second hospital day, and grossly edematous and necrotic muscle was found.
The medial (adductor) compartments were soft when palpated, and they were not
opened.
The clinical course continued to deteriorate over the next twenty-four
hours, requiring admission to the intensive-care unit for the management of a
systemic inflammatory response syndrome—a proinflammatory state that
manifested as tachycardia, tachypnea, hypotension, hypoperfusion, oliguria,
and the need for further aggressive volume resuscitation and pharmacologic
vasopressor support. The myonecrosis continued to worsen
(Table II), and there was tense
swelling and rapidly increasing pain in the calves as well as pain with
passive stretch, particularly with dorsiflexion of the feet and toes. In
addition, the patient reported decreased sensation over the dorsal and plantar
surfaces of both feet. The diagnosis of compartment syndrome was made on the
basis of the history and examination alone, without the use of invasive
pressure monitoring. The patient underwent emergent bilateral four-compartment
fasciotomies through a single lateral incision on the third hospital day.
The thirty-three-day hospital course included nine additional operative
interventions for extensive débridement of devitalized muscle from the
thighs and three surgical débridements of the legs. The fasciotomy
incisions on the legs were treated with delayed primary closure on the sixth
postoperative day. The thigh incisions could not be closed because of
excessive skin tension, and they were treated definitively with
vacuum-assisted wound closure and wet-to-dry dressings.
Heterotopic ossification was initially identified on radiographs of the
thighs one month after the initial presentation. The ectopic bone became
extensive enough (Fig. 1) to
cause skin ulceration on the left and a partial wound dehiscence on the right,
despite administration of indomethacin (25 mg, three times daily). At eight
months postoperatively, the functional deficits were worse on the left, with
only 40° (10° to 50°) of knee motion compared with 70°
(10° to 80°) on the right. Excision of heterotopic bone, followed by a
single dose of radiation therapy (700 cGy) as additional prophylaxis, was
performed eight months postoperatively on the left and ten months
postoperatively on the right (Fig.
2).
At the time of final follow-up, fourteen months after the initial
presentation, the wounds had healed and the functional outcome was
satisfactory. The arc of knee motion had improved to 100° (0° to
100°) on the left and 95° (5° to 100°) on the right. Despite
some functional weakness, the lower-extremity strength was normal on manual
motor testing.
This report describes a worrisome association between intense exercise and
creatine supplementation and the development of compartment syndrome in the
lower extremity. Furthermore, it describes the development of massive
heterotopic ossification as a complication of delayed treatment of acute
compartment syndrome necessitating additional medical as well as surgical
intervention.
Exercise-Induced Compartment Syndrome
Compartment syndrome of the thigh is believed to be rare because of the
large volume of the thigh compartment and its contiguous relationship with the
muscular compartments of the hip and gluteal
region1. Knowledge
of its association with exercise is important, as this syndrome occurs more
commonly in association with skeletal trauma, vascular injury, or prolonged
compression. It appears that creatine supplements are frequently used by
patients in whom exercise-induced compartment syndrome
develops3,5,6.
As a naturally occurring substance synthesized primarily within the liver,
kidneys, and pancreas, it is used by the body as a substrate for adenosine
triphosphate (ATP)
synthesis7. At the
recommended maintenance dose, which is 2 to 5 g/day for a 70-kg
patient8, creatine
supplementation has been shown to increase total body weight by an average of
0.8 kg over a period of eight days, solely as a result of an increase in total
body water and intracellular fluid
volume9. While, to
our knowledge, no studies have directly implicated creatine supplementation as
a mechanism affecting muscle compartment pressures, human trials have shown
that ingested creatine is transported into muscle tissue, probably increasing
muscle volume and water
content6. Increasing
muscle volume within a closed fascial space may predispose a patient to the
development of compartment syndrome.
Heterotopic Ossification Secondary to Delayed Treatment of
Compartment Syndrome
The formation of heterotopic bone as a consequence of compartment syndrome
has not been previously described, to our knowledge. However, we believe that
the etiology of the heterotopic bone formation in our patient was probably
multifactorial. The initial muscular necrosis resulting from the delay in the
treatment of the acute compartment syndrome, combined with repeated muscular
injury secondary to multiple surgical débridements, represents the most
likely etiology of the heterotopic bone formation in this patient. Animal
studies support this suggested etiology by implicating an interaction between
ischemic muscle and injured periosteum as a mechanism that can induce
ossification in the surrounding
muscle10,11.
Trauma, unquestionably, is the most common condition associated with the
formation of heterotopic
bone12. Although
the exact etiology remains unknown, it is believed that muscle injury
sufficient to cause proliferative repair is essential to the formation of
heterotopic bone13.
Described risk factors include scars, burns, paraplegia, cerebral injury,
neurological disorders (poliomyelitis), and
spondyloarthropathies14.
In addition, prolonged immobilization, stasis, edema, and swelling, all of
which are common in patients treated surgically for acute compartment
syndrome, are factors that encourage ectopic bone formation.
Techniques utilized to prevent formation of heterotopic bone are diverse
and include pharmacotherapy, radiation therapy, and surgical intervention or a
combination of these treatments. Indomethacin has been demonstrated to prevent
the formation of heterotopic bone, albeit with adverse gastrointestinal
effects and the potential for nephrotoxicity in patients with tenuous
intravascular
volume14,15.
Radiation therapy is an alternative modality for prevention of heterotopic
bone, although the cost and the exposure to ionizing radiation are inherent
disadvantages16-18.
Finally, it is thought that early resection of necrotic muscle during the
initial traumatic event may be effective in preventing heterotopic
ossification in high-risk
patients19. This
may be particularly relevant in patients treated with fasciotomy for delayed
presentation of acute compartment syndrome, in whom repeat débridement
of necrotic muscle is common.
The management of compartment syndrome requires early diagnosis and prompt
decompression and typically results in an uncomplicated
recovery20. To
facilitate this goal, it is essential to understand the relationship between
exercise and the development of acute compartment syndrome. Similarly,
recognizing heterotopic bone as a potential complication associated with the
delayed treatment of acute compartment syndrome is important, as close
follow-up, medical treatment, and additional surgical intervention may be
required. ?