Introduction

Compartment syndrome is a condition in which increased interstitial pressure within a closed myofascial area leads to decreased tissue perfusion and subsequent tissue necrosis [1, 2]. It is an acute limb and life-threatening emergency [2, 3]. The most commonly involved areas are the lower and upper extremities and the abdomen [4, 5]. Gluteal Compartment Syndrome (GCS) is rare and thus associated with a low index of suspicion among clinicians; hence, it is often missed or diagnosed late [2]. In the gluteal region, any of the three fascial compartments can be affected: the anterior tensor fascia lata (TFL), the gluteus medius and minimus, and the posterior gluteus maximus [2, 6].

There is a paucity of literature when it comes to the risk factors and etiologies associated with GCS. Other small case series seem to suggest that GCS arises from trauma, prolonged immobilization secondary to surgery, or substance abuse [3, 6,7,8]. Similar to compartment syndrome of the leg, patients with GCS typically present with severe pain that is, sometimes but not always, associated with swelling, erythema, and tenderness to the gluteal region. This often progresses to paresthesia and paralysis of the sciatic nerve. The cornerstone of treatment is prompt diagnosis and early decompressive fasciotomy [9,10,11] to avoid debilitating complications such as permanent paralysis and/or muscle tissue loss [12, 13], rhabdomyolysis often leading to myoglobinuria and acute renal failure, and potentially death [14].

GCS is a devastating condition that appears to be more common and associated with more complications than previously acknowledged, yet its atypical presentation may limit familiarity with the diagnosis and lead to a clinical deficit when comparing our preparedness for the diagnosis of GCS versus the debilitating results it can have to the patient. To our knowledge, there is only one case series reporting GCS in ≥ 5 patients (n = 13) at a tertiary care level 1 trauma center from 2007 to 2018 [15]. Yet, it focused less on the etiology and risk factors for GCS and more on the diagnosis and management. It did not include race, medical comorbidities, and laboratory work-up, and demonstrated a significantly lower survival rate when compared to prior studies. Therefore, the purpose of our study is to (1) describe the presentation, symptoms and etiology of GCS and (2) evaluate the survival rate at our tertiary care institution.

Methods

Study design

This is an IRB-approved retrospective case series of patients diagnosed with GCS at a Tertiary Academic Level I Trauma Center. All patients who were identified by the Current Procedural Terminology (CPT) codes for undergoing thigh and/or gluteal fasciotomies (CPTs: 27,025, 27,027, 27,057, 27,496, 27,497, 27,498, 27,499) between January 2015 and December 2022 were included in this study. All records in which operative notes indicate that no fasciotomy was performed or that fasciotomies were done exclusively for thigh compartments were excluded (Fig. 1).

Fig. 1
figure 1

Flow diagram outlining the identification of patients with GCS at our tertiary care level 1 trauma center

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Data collection

Charts were reviewed to extract data on patient demographics (age, sex, race, BMI), presenting symptoms (trauma, fracture, mechanism of injury, associated symptoms), comorbidities and risk factors (diabetes, hypertension, congestive heart failure, current anticoagulation, substance use), preoperative lab values, intraoperative findings, and postoperative outcomes.

Statistical analysis

Descriptive statistics were presented as the mean ± standard deviation (SD) for continuous variables and N (%) for categorical variables. Analysis was conducted using Stata Statistical Software: Release 17 (StataCorp. 2021; College Station, TX).

Results

Patient demographics

12 (85.7%) out of 14 patients were male, with an average age of 39.4 ± 13 years. Mean BMI was near normal (25.1 ± 4.1 kg/m2). 7 (50%) patients were White, 4 (28.6%) patients were Black/African American, 2 (14.3%) patients were Hispanic, and 1 (7.1%) patient was Asian. These findings are summarized in Table 1.

Table 1 Patients demographics
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Presenting symptoms, comorbidities, and risk factors

Out of the 14 patients, only 2 patients presented to the hospital classified as “Trauma”. 2 patients presented with one fracture each, a proximal femur fracture and a nondisplaced first rib fracture. 1 patient presented with three fractures including a displaced, comminuted femoral shaft fracture, iliac bone fracture, and inferior pubic ramus fracture. Mechanisms of injury were categorized as ballistic (1), “found-down” (6), ground-level fall (1), and iatrogenic (6) causes. 2 patients were intubated at the time of evaluation and diagnosis. 5 (35.7%) patients had an associated neurovascular injury at the time of GCS diagnosis, and 7 patients demonstrated some degree of sciatic nerve palsy on physical exam. 9 (64.3%) patients reported drug use, with 8 (88.9%) of them reporting polysubstance use. The most commonly reported drugs were cocaine (7) and heroin (7). 7 patients reported alcohol use. 3 (21.4%) patients had hypertension and 1 patient had insulin-dependent diabetes mellitus. None of the patients had COPD or CHF. 2 (14.3%) patients were receiving anticoagulation at the time of diagnosis (1 for acute exacerbation of antiphospholipid syndrome, and 1 for acute myocardial infarction). These findings are summarized in Table 2 and Supplementary Table 1.

Table 2 Patients clinical characteristics
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Preoperative lab values

Standard lab values were recorded at the time of diagnosis of compartment syndrome. Mean hemoglobin (Hgb) was found to be 11.7 ± 4 g/dL, with 5 patients recording a value less than 10 g/dL. Conversely, creatine kinase (CK) was elevated in all 14 patients (49,617 ± 60,068 units/L), and lactate levels were elevated in 9 patients with a mean of 2.8 ± 1.6 mmol/L. Other standard lab values included international normalized ratio (INR) (1.1 ± 0.14), white blood cells (WBC) (14.9 ± 7.3 count/L × 103), platelets (239 ± 123 × 109/L) and creatinine (2.5 ± 1.2 mg/dL). These findings are summarized in Table 3.

Table 3 Preoperative findings
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Intraoperative findings

All patients received gluteal compartment fasciotomy. Of those, 8 (57.1%) patients were also diagnosed with concomitant thigh compartment syndrome and underwent thigh compartment release. Intraoperatively, the mean estimated blood loss (EBL) was 762 ± 1886 mL. 6 (42.9%) patients demonstrated intraoperative evidence of gluteal compartment hematomas, and 13 patients had non-viable muscle requiring debridement. These findings are summarized in Table 4.

Table 4 Intraoperative findings
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Postoperative outcomes

Postoperatively, the overall hospital length of stay was 25 ± 22 days, with 13 patients requiring a stay of at least 7 days. The mean intensive care unit (ICU) length of stay was 11 ± 22 days. Some complications developed post-operatively: pneumonia (7.1%), deep vein thrombosis (7.1%), and myocardial infarction (14.3%). None of the patients developed a soft tissue infection, sepsis, or a postoperative compartment syndrome. 2 patients (14.3%) died during hospital admission. None of the deaths were directly related to the GCS; one patient died from COVID-induced acute respiratory distress and the other died from extensive injuries following gunshot wounds (Supplemental Table 2). The remaining 12 patients all required discharge to advanced rehabilitation facilities. These findings are summarized in Table 5.

Table 5 Postoperative findings
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Discussion

To our knowledge, this is the second and currently largest tertiary-care-center retrospective case series to assess etiology and clinical presentation with a focus on outcomes of patients with GCS. Patients with GCS were more likely to present as young to middle-aged males, with a history of polysubstance use, minimal chronic medical comorbidities, and were either found-down or had experienced an iatrogenic injury (Fig. 2). Short-term outcomes were generally poor with many patients experiencing muscle necrosis, developing nerve palsy, and requiring advanced care for an extended period of time (Fig. 2).

Fig. 2
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Graph summarizing the number of GCS patients per significant characteristic

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In terms of patient demographics, there seems to be a consensus around the sex and average age of patients presenting with GCS, where the literature and our study suggest that it is likely to occur in young to middle-aged males [2, 6, 15]. In contrast to previous studies where GCS patients were mostly obese with a mean BMI of around 41 kg/m2 [2, 15], the mean BMI of our cohort was near normal. While our study is the first to report patients’ race, there was an equal number of white and non-white GCS patients in our study; the rarity of GCS and the subsequently small sample size limit our ability to draw any conclusions regarding racial disparities in GCS patients at this stage.

Interestingly, at presentation, GCS etiology was most commonly attributed to iatrogenic causes or to the classic “found-down” patient, despite similar outcomes (Supplementary Tables 1 and 2). The former is associated with the inadvertently inherent immobilization of the gluteal compartment area intraoperatively and the stress on the muscles and surrounding neurovascular structures, both of which can lead to muscle ischemia and increased compartmental pressure [16, 17]. Only three patients presented to our hospital with one or more fractures and only two of them were classified as “traumas”. However, the literature suggests that GCS is typically associated with fractures [4, 6] and is often caused by direct trauma [13]. Alike published findings, most patients had a history of alcohol and polysubstance use, notably cocaine and heroin. This may contribute to longer times of immobilization, whether in the found-down or post-operative patient, with an extended duration of direct compression and pressure on the dependent structures [18, 19]. The wide variety of mechanisms of injury behind GCS might have been under-reported or under-represented. It may be too early to confirm a specific etiology for GCS, and physicians should keep a high suspicion for such a potentially devastating condition given the various presentations. Moreover, this is the first study to report medical comorbidities in GCS patients. Surprisingly, most patients had no medical comorbidities and were not receiving any prescribed medications or anticoagulation at the time of diagnosis, suggesting that GCS can affect otherwise healthy individuals. On physical examination, 50% of patients had some degree of sciatic nerve palsy resulting from compression—an expected finding, given the anatomical proximity of the sciatic nerve to the gluteal region, which has been demonstrated in multiple case reports and reviews [4, 6, 8, 13, 15, 17, 18]. Looking at laboratory findings, low Hgb, and high levels of CK and lactate were more common than not. While previous studies have reported similarly high CK levels [3, 15], very few documented other lab values such as Hgb, or INR, WBCs, platelets, and creatinine—all four of which were within normal range in this study. Further investigation is needed to be able to draw conclusions regarding lab findings in the GCS patient population.

Intra-operatively, all patients received gluteal compartment fasciotomy. While this is in line with the classic management of compartment syndrome [2, 3], it contrasts the most recent case series by Adib et al. where fasciotomy was restricted to non-crush injury patients who were neurologically intact at presentation or who developed neurologic deficits despite medical management. Their recommendation was cautiously determined following their 69% inpatient survival rate [15]—a survival significantly lower than the 85.7% found in our tertiary care center or the 95% reported in all previously published cases. Indeed, while analyzing their patients’ subset or the entire cohort across published studies, mortality was not statistically significantly lower for those who had undergone fasciotomy [15]. Other studies suggest that a delay in diagnosis would also lead to elective medical treatment, as the potential risks associated with fasciotomy may outweigh the benefits [15, 20, 21]. This contrasts with Lawrence et al. who reported immediate clinical improvement in a patient with neurologic symptoms after a 56-h delayed fasciotomy [11]. This suggests that fasciotomy may play a crucial role in the management of GCS but needs to be chosen on a case-by-case basis, after careful assessment. This is the first study to report mean EBL, and it was found to be significantly elevated. This is not surprising given that almost half of our GCS patients demonstrated evidence of gluteal compartment hematomas and almost all had non-viable muscle tissue requiring debridement.

Post-operatively, all patients experienced extended ICU stays and, while only two died during hospital admission, the remaining 12 required advanced rehabilitation. Unfortunately, the follow-up was not long enough to allow for a more critical assessment of outcomes following rehabilitation sessions. The importance attributed to survival in contrast to post-operative outcomes is a premise that warrants further investigation.

Finally, it is important to note that the two largest case series of this condition shared the same geographical location. Yet, while the overall findings were similar across cohorts, there was a spectrum of etiologies and the survival rate significantly differed between the two institutions. As both are Level 1 trauma tertiary care centers, this finding refutes the previously published assumption which suggests that the nature of the center attracted more complex cases resulting in outcomes discrepancies with the published literature. This might be explained by the difference in GCS management, where our GCS patients all had fasciotomies done. Research is still needed to better elucidate which management optimizes both outcomes and survival.

GCS remains particularly rare and its infrequent and still inconsistent presentation in the literature may complicate accurate and time-efficient diagnosis. Physicians should keep a high suspicion for this potentially devastating condition to avoid the detrimental effects that a delayed or missed diagnosis can have on the patient, from morbidity with longer hospital stays and rehabilitation programs to mortality. Further investigation towards understanding this particular patient population can improve diagnosis and treatment.