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Keywords:

  • arterial intervention;
  • angiogram;
  • embolization;
  • haemorrhage;
  • pseudoaneurysm

We present an unusual case of pseudoaneurysm of the superior gluteal artery causing gluteal compartment syndrome after bone marrow biopsy of the iliac crest performed for the evaluation of thrombocytopenia.

A 55-year-old man presented with an episode of ‘collapse’ 1 h after a bone marrow aspiration from his right iliac crest. There was no loss of consciousness and the primary complaint was that of pain in the buttock at the site of the bone marrow biopsy, ‘shooting’ pains to the foot, which increased with movement and a feeling of ‘tingling’ in his foot. On examination, there was no bruising, the pulses were intact and there was full range of movement in the limb with no focal neurology. Observations were normal.

Initially the patient was treated with analgesia and observation. Blood tests demonstrated a mild anaemia (Hb 114 g/l) and normal coagulation screen.

A computed tomography (CT) scan was performed in portal-venous phase. This demonstrated active extravasation of contrast into the gluteal compartment with small haematoma and mass effect on the sciatic nerve, suggesting a compression-induced neuropathy.

A diagnosis of iatrogenic pseudoaneurysm of the superior gluteal artery causing gluteal compartment syndrome was made.

The case was discussed with a Consultant Interventional Radiologist and the patient was taken into the vascular suite, and a formal angiogram performed with a view to embolization.

At angiogram, the pseudoaneurysm was easily visualized, however, selected catheterization demonstrated that it arose at the division of the artery into five distinct branches (Fig 1).

image

Figure 1. Angiogram with catheterization of the superior gluteal artery showing pseudoaneurysm.

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Coil embolization was performed to good effect using microcoils (Fig 2).

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Figure 2. Coil embolization.

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A duplex ultrasound scan was performed the following day and showed no flow within the pseudoaneurysm.

The patient was allowed home 24 h after embolization, walking with the aid of a frame and with significant improvement of his sciatica symptoms, but persistent, lesser symptoms of buttock pain.

When attending the Interventional Radiology out-patient clinic 6 weeks later, the patient walked with the aid of a stick and had complete resolution of his sciatica with no foot drop. The patient was discharged from clinic.

Bone marrow aspiration is used to diagnose various haematological disorders and to estimate prognosis and response to therapy (Sarigianni et al, 2011). Associated complications are rare (0·05%) (Bain, 2006). The commonest and most serious complication is haemorrhage (Sarigianni et al, 2011). Local haematoma, retroperitoneal haemorrhage and nerve damage have been described in the literature (Arellano-Rodrigo et al, 2004; Bain, 2006; Sarigianni et al, 2011), as has injury to the gluteal artery specifically (Roth & Newman, 2002; Bain, 2006). Gluteal compartment syndrome (GCS) is a rare, often unrecognized syndrome seldom reported in the literature (Mustafa et al, 2009).

Compartment syndrome is a well-recognized phenomenon, most commonly encountered in the lower leg and forearm. Elevated intracompartmental pressures compromise blood flow to involved muscles and nerves resulting in ischaemia, cellular damage and oedema. This leads to further increases in compartmental pressure, and subsequent worsening of the perfusion deficit and consequent nerve and muscle damage. The gluteal region is not always thought of as being composed of compartments, however, three nondistensible osseofacial boundaries do exist (Bleicher et al, 1997; Roth & Newman, 2002).

Gluteal compartment syndrome as a result of trauma, drug/alcohol use and immobilization have been reported (Kumar et al, 2007; Mustafa et al, 2009). Sporadic reports of iatrogenic, post-surgical injuries can also be found (Bleicher et al, 1997; Kumar et al, 2007; Ge et al, 2010; Taylor et al, 2011).

Gluteal artery pseudoaneurysms are exceedingly uncommon but should be considered in the patient with buttock pain and sciatic nerve palsy following iliac crest bone marrow biopsy.

The documented risk factors associated with haemorrhage following bone marrow biopsy are: myeloproliferative neoplasm, aspirin or warfarin therapy, disseminated intravascular coagulation and obesity (Bain, 2006; Sarigianni et al, 2011).

As with any case of compartment syndrome, expedient therapeutic measures are critical to salvaging neuromuscular function (Roth & Newman, 2002). Treatment is traditionally surgical, often requiring multiple fasciotomies, with which there is recognized morbidity (Ge et al, 2010; Taylor et al, 2011). The rising profile and expertise within Interventional Radiology and Endovascular techniques advent a new era - allowing a purely endovascular, or a combined approach to these cases.

The development of a gluteal artery pseudoaneurysm as a complication of bone marrow biopsy is an extremely rare phenomenon with only a handful of cases being documented in the literature (Chamisa, 2007; Ge et al, 2010). However, it is a serious complication with a considerable morbidity (Ge et al, 2010). Previously documented cases have presented at day 6, day 17 and 6 weeks following bone marrow biopsy with symptoms of buttock pain, neuropathic leg pain and abdominal pain respectively. None were accurately diagnosed acutely at initial presentation.

In 2007, Chamisa described a 29-year-old man with with significant cardiac abnormalities who presented with sudden onset abdominal pain, vomiting, hypovolaemic shock, and generalized peritonitis one hour following a bone marrow biopsy performed for the evaluation of thrombocytopenia. CT demonstrated a left superior gluteal artery pseudoaneurysm, and following unsuccessful coil embolization, he developed abdominal compartment syndrome and died 2 d later as a result of fatal retroperitoneal haemorrhage. Surgical intervention was not undertaken due to the patient's cardiac risk.

The patient in our report had very rapid symptom onset, with the patient attending the Emergency Department, having not managed to leave the hospital after his bone marrow biopsy. In view of this acute presentation, a CT was considered early on and subsequently the pseudoaneurysm was diagnosed and treated in a timely fashion. Because of this, we were able to successfully treat by endovascular means alone – avoiding fasciotomies and allowing early restoration of function with only few residual symptoms at 6 weeks. It looks promising that these residual symptoms will also resolve with time.

This case serves as reminder that although generally considered safe, there are significant risks to bone marrow biopsy and unless these adverse outcomes are considered, they can easily be overlooked, delaying diagnosis and definitive treatment.

Author contributions

  1. Top of page
  2. Author contributions
  3. Conflict of interest
  4. References

C Sullivan performed the literature review, obtained and edited the images and wrote and edited the report.

M Regi performed the initial CT scan and subsequent arterial embolization. He also critically reviewed and approved the final version of the paper.

Conflict of interest

  1. Top of page
  2. Author contributions
  3. Conflict of interest
  4. References

The authors declare no conflict of interest.

References

  1. Top of page
  2. Author contributions
  3. Conflict of interest
  4. References
  • Arellano-Rodrigo, E., Real, M.I., Muntañola, A., Burrel, M., Rozman, M., Fraire, G.V. & Cervantes, F. (2004) Successful treatment by selective arterial embolization of severe retroperitoneal hemorrhage secondary to bone marrow biopsy in post-polycythemic myelofibrosis. Annals of Hematology, 83, 6770.
  • Bain, B.J. (2006) Morbidity associated with bone marrow aspiration and trephine biopsy–a review of UK data for 2004. Haematologica, 91, 12931294.
  • Bleicher, R.J., Sherman, H.F. & Latenser, B.A. (1997) Bilateral gluteal compartment syndrome. Journal of Trauma, 42, 118122.
  • Chamisa, I. (2007) Fatal vascular retroperitoneal injury following bone marrow biopsy. South African Medical Journal, 97, 246.
  • Ge, P.S., Ng, G., Ishaque, B.M., Gelabert, H. & de Virgilio, C. (2010) Iatrogenic pseudoaneurysm of the superior gluteal artery presenting as pelvic mass with foot drop and sciatica: case report and review of literature. Vascular and Endovascular Surgery, 44, 6468.
  • Kumar, V., Saeed, K., Panagopoulos, A. & Parker, P.J. (2007) Gluteal compartment syndrome following joint arthroplasty under epidural anaesthesia: a report of 4 cases. Journal of Orthopaedic Surgery (Hong Kong), 15, 113117.
  • Mustafa, N.M., Hyun, A., Kumar, J.S. & Yekkirala, L. (2009) Gluteal compartment syndrome: a case report. Cases Journal, 2, 190.
  • Roth, J.S. & Newman, E.C. (2002) Gluteal compartment syndrome and sciatica after bone marrow biopsy: a case report and review of the literature. American Surgeons, 68, 791794.
  • Sarigianni, M., Vlachaki, E., Chissan, S., Klonizakis, F., Vetsiou, E., Anastasiadou, K.I., Ionnidou-Papagiannaki, E. & Klonizakis, I. (2011) Haematoma caused by bone marrow aspiration and trephine biopsy. Haematological Reports, 3, e2578e2579.
  • Taylor, B.C., Dimitris, C., Tancevski, A. & Tran, J.L. (2011) Gluteal compartment syndrome and superior gluteal artery injury as a result of simple hip dislocation: a case report. The Iowa Orthopaedic Journal, 31, 181186.