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Delayed Presentation of Gluteal Compartment Syndrome due to Pseudoaneurysm Rupture

A Case Report

Braswell, Matthew J. MD1; Anderson, Ashley MD2; Donohue, Michael MD2; DiVito, Michelle C. MD2; White, Paul W. MD2; Wagner, Scott C. MD2

doi: 10.2106/JBJS.CC.18.00346
Case Reports
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Disclosures

Case: We present a 26-year-old active duty man who sustained a coccygeal fracture and gluteus maximus hematoma after a fall from height during training. The patient returned 3 weeks postinjury with symptoms and physical examination findings concerning for gluteal compartment syndrome. An expanding gluteal hematoma was confirmed on imaging and the patient was taken to the operating room for emergent evacuation and endovascular hemostasis.

Conclusions: This case of gluteal compartment syndrome is a unique contribution to the literature with respect to the specific vascular injury observed and the delayed presentation of gluteal compartment syndrome.

1Department of Orthopaedic Surgery, Carolinas Medical Center, Charlotte, North Carolina

2Department of Orthopaedic Surgery, Walter Reed National Military Medical Center, Bethesda, Maryland

E-mail address for M.J. Braswell: Braswellmj@gmail.com

Investigation performed at Walter Reed National Military Medical Center, Bethesda, Maryland

Disclosure: The Disclosure of Potential Conflicts of Interest forms are provided with the online version of the article (http://links.lww.com/JBJSCC/A838).

Compartment syndrome is a sequela of trauma in which interstitial pressure in an osseofascial compartment exceeds the perfusion pressure of the relevant capillary vasculature, resulting in compartment ischemia, cellular hypoxia, and eventually tissue death1. The syndrome usually presents with pain out of proportion to known injuries, exacerbated by passive stretch motions, and resistance to opiate pain medications1. The affected compartment is often palpably tense on physical examination. Vascular signs, such as pallor, a cold, or pulseless extremity, and neurological signs, such as paresthesias and paralysis, can be late findings of the syndrome1. The use of a wick catheter to monitor compartment pressures has been described2, with compartment pressure >30 mm Hg or within 30 mm Hg of diastolic blood pressure considered suggestive for compartment syndrome. Although many compartment syndromes result from intracompartmental fractures or vascular injuries, there is an extracompartmental subtype caused by constrictive dressings, prolonged patient positioning, and circumferential burns1.

Compartment syndrome most often occurs in the distal extremities, but has been described in other anatomical areas. Gluteal compartment syndrome is one of the rarest forms3, and when it does occur, it most commonly develops secondary to prolonged immobilization after a surgical operation or drug/alcohol intoxication2,4. Cases have been described following lumbar spine surgery5, total hip arthroplasty6, total knee arthroplasty7, posterior cruciate ligament repair8 due to prolonged immobilization, as well as following bone biopsy9 and intramuscular injection10. Major and minor trauma are additional causes. The first gluteal compartment syndrome case described was in a 30-year-old physician who fell from standing height onto a tennis ball, though his symptoms resolved spontaneously2. Delayed presentation of gluteal compartment syndrome after trauma has never been described in the literature.

The gluteal compartment is composed of 3 separate subcompartments: the gluteus maximus compartment, the gluteus medius and minimus compartment, and the tensor fascia lata compartment2. Deep to the gluteal compartment runs the sciatic nerve, as do the superior and inferior gluteal nerves11. The internal iliac branches exit the greater sciatic foramen as well, specifically the superior and inferior gluteal arteries. The superior gluteal artery (SGA) travels in close proximity to the sciatic notch, supplies the gluteus medius, before splitting along the gluteus minimus into the deep superior SGA, deep inferior SGA, and superficial SGA. These branches then anastomose with the lateral femoral circumflex artery11. The inferior gluteal artery (IGA) exits the greater sciatic foramen and travels to supply the gluteus maximus and continues inferiorly to supply the distal sciatic nerve3,11. During operative management, all 3 subcompartments of the gluteal compartment must be released to allow for perfusion of all muscles and the sciatic nerve by the IGA and SGA as described3.

The patient was informed that data concerning the case would be submitted for publication, and he provided consent.

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Case Report

We present the case of a 26-year-old active duty man who sustained a displaced coccygeal fracture (Fig. 1) and right gluteus maximus hematoma after a fall from height during a training exercise. After 3 weeks of uncomplicated recovery and progressive mobilization, he re-presented to the emergency room with worsening pain and increasing narcotic requirements. On physical examination, the patient was found to have a tense right gluteal compartment and was uncomfortable in any position other than prone. He had no neurological deficits and was hemodynamically stable. The patient refused a new computed tomography scan, so magnetic resonance images of the pelvis were obtained (Figs. 2 and 3). The images demonstrated large heterogeneous fluid collections concerning for hematoma expansion involving the right gluteus maximus muscle bodies. Based on the symptoms, physical examination, and imaging findings, there was clinical concern for an evolving compartment syndrome.

Fig. 1

Fig. 1

Fig. 2

Fig. 2

Fig. 3

Fig. 3

The decision was made at that time to proceed with gluteal compartment fasciotomies and hematoma evacuation. An extensile Kocher-Langenbeck posterior approach was made down to the superficial fascia of the tensor fascia lata (Fig. 4). Once the gluteal fascia was incised, a large coagulated hematoma was encountered. As the hematoma was evacuated, severe hemorrhage was encountered at the level of the sciatic notch that was not well controlled with ligation attempts. Emergent hemostasis/fluid resuscitation protocols were initiated, although the hemorrhage was adequately controlled with packing and manual compression. Vascular surgery and a senior orthopaedic trauma specialist were consulted intraoperatively with concern for superior/IGA injuries, but despite several attempts, adequate hemostasis could not be achieved through open ligation. Interventional radiology was then consulted intraoperatively for emergent angiography. The patient was transferred to the interventional radiology suite while the surgical team held manual pressure on the packed wound. Angiography revealed injuries to both the SGA and IGA with pseudoaneurysm formation. Embolization with coiling of both gluteal arteries was successfully performed (Figs. 5 and 6). Once completed, hemostasis was achieved, and the surgical wound was irrigated and closed.

Fig. 4

Fig. 4

Fig. 5

Fig. 5

Fig. 6

Fig. 6

The patient's postoperative course was uncomplicated. He recovered quickly from a hemodynamic and physiologic perspective. When seen in clinic for postoperative follow-up, the patient demonstrated no functional or neurologic deficits on examination and returned to his active duty station with no restrictions.

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Discussion

Our case contributes to growing evidence for the incidence of gluteal compartment syndrome in varying energy levels of trauma. On review of the literature, there have been 3 cases of gluteal compartment syndrome in high energy trauma in which there were associated pelvic injuries, 2 with pelvic fractures and documented SGA ruptures12,13 and one with an anterior-posterior compression injury resulting in sacroiliac subluxation and pubic symphysis diastasis in which vascular injury was not documented14. An additional case of high energy trauma resulting in compartment syndrome was reported in 1990 by Brumback. Although the patient had no pelvic fractures, the elevated gluteal compartment pressure was a result of an SGA injury15. Exactly one case of high energy trauma and gluteal compartment syndrome has been described in which SGA rupture occurred after a simple hip dislocation in the absence of any pelvic ring fractures16.

There have been cases of gluteal compartment syndrome in lower energy trauma. In one such case, a patient fell from standing height and acutely presented with SGA rupture, resulting in gluteal compartment syndrome3. Rocos and Ward17 described a patient who sustained a fall from a ladder resulting in no acute fractures, who then developed a gluteal hematoma and compartment syndrome secondary to a lacerated IGA. Excepting the current case report, this patient is the only other documented case of an IGA injury resulting in gluteal compartment syndrome. All cases of gluteal compartment syndrome following trauma that have been documented were cases in which the syndrome developed acutely, immediately following trauma, which makes the delay in presentation of our case highly unique.

Combined endovascular embolization and open fasciotomy have been described in several other cases for management of gluteal compartment syndrome. However, the currently presented case is unusual, in that both the IGA and SGA were ruptured in a low energy trauma incident, and despite the severity of the vascular insult, development of acute compartment syndrome did not occur for 3 weeks postinjury. It is possible that increased activity by the patient led to repeat vascular injury, which in turn led to the formation of a new gluteal hematoma and a spike in intracompartmental pressure. In addition, it is likely that the vascular injuries at the time of trauma led to formation of the multiple pseudoaneurysms noted at the time of endovascular hemostasis (Fig. 5). These pseudoaneurysms would lend the superior and inferior gluteal arteries vulnerable to repeat injury, especially as the patient increased his activity level. Regardless of the pathogenesis, the delayed presentation is unprecedented in the literature and serves as a warning for clinicians to suspect compartment syndrome even with a delayed presentation and in the setting of trauma of varying levels.

Clinicians must maintain high suspicion for compartment syndrome, and when necessary fasciotomies and endovascular embolization must be performed emergently. Delays in care for compartment syndrome can have disastrous consequences. The resulting muscle and nerve necrosis can initiate a pathway of systemic dysregulation including acidosis, rhabdomyolysis, and myoglobinuria, and eventually acute kidney failure and death18. Whiteside and Hirada demonstrated that ischemia for 4 or more hours can induce irreparable muscle damage19; however, muscle ischemia and death can occur before this described timeline, as has been specifically noted intraoperatively during a case of gluteal compartment syndrome3. In gluteal compartment syndrome specifically, delayed diagnosis and/or management causes ischemia and death of the muscles discussed and can result in sciatic nerve damage. Damage to the sciatic nerve can worsen the long-term leg weakness from loss of viable muscle tissue, resulting in significant loss in ability to perform activities of daily living14,17. The sciatic nerve damage can also cause paresthesia or hyperesthesia that affects the quality of life of the patient18. The life-threatening and debilitating consequences of gluteal compartment syndrome are made clear by the cases in the literature, as is the incidence of the pathology and the need for consistent and aggressive management by providers.

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References

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