Acute Compartment Syndrome Following Allograft-Prosthetic Composite Reverse Shoulder Arthroplasty for Osteosarcoma of the Proximal Humerus: A Case Report

Acute compartment syndrome (ACS) is a surgical emergency. It occurs when there is an increase in pressure within a closed fascial compartment leading to decreased circulation and eventual ischemia and irreversible necrosis.4Giai Via A. Fioruzzi A. Randelli F. Diagnosis and Management of Snapping Hip Syndrome: A Comprehensive Review of Literature.Rheumatol Curr Res. 2017; 7https://doi.org/10.4172/2161-1149.1000228Crossref Google Scholar,15Torlincasi AM, Waseem M. Compartment Syndrome, Extremity. Treasure Island, FL StatPearls Publishing No doi no ISBNGoogle Scholar Although it is considered a clinical diagnosis, measurements of intracompartmental pressures > 30mmHg is used as a threshold to aid in diagnosis.4Giai Via A. Fioruzzi A. Randelli F. Diagnosis and Management of Snapping Hip Syndrome: A Comprehensive Review of Literature.Rheumatol Curr Res. 2017; 7https://doi.org/10.4172/2161-1149.1000228Crossref Google Scholar,11Schwartz J.T. Brumback R.J. Lakatos R. Poka A. Bathon G.H. Burgess A.R. Acute compartment syndrome of the thigh. A spectrum of injury.J Bone Joint Surg Am. 1989 Mar; 71: 392-400Crossref PubMed Scopus (187) Google Scholar Without prompt diagnosis and surgical intervention, ACS can result in severe morbidity or even mortality.11Schwartz J.T. Brumback R.J. Lakatos R. Poka A. Bathon G.H. Burgess A.R. Acute compartment syndrome of the thigh. A spectrum of injury.J Bone Joint Surg Am. 1989 Mar; 71: 392-400Crossref PubMed Scopus (187) Google Scholar Although ACS more commonly affects the lower extremity, ACS of the upper extremity can result in profound disability for patients.8Maeckelbergh L. Colen S. Anné L. Upper arm compartment syndrome: a case report and review of the literature.Orthop Surg. 2013 Aug; 5: 229-232https://doi.org/10.1111/os.12054Crossref PubMed Scopus (13) Google Scholar Fractures are the most common causes of ACS of the upper extremity, while use of anticoagulation therapy, hematoma formation, pectoralis muscle tendon rupture, biceps tendon rupture, and prolonged lateral decubitus position have been reported as etiologies as well. 3Fung D.A. Frey S. Grossman R.B. Rare case of upper arm compartment syndrome following biceps tendon rupture.Orthopedics. 2008 May; 31: 494https://doi.org/10.3928/01477447-20080501-09Crossref PubMed Scopus (15) Google Scholar,9Meshram P. Joseph J. Zhou Y. McFarland E.G. Acute compartment syndrome caused by hematoma after shoulder surgery: a case series.J Shoulder Elbow Surg. 2021 Jun; 30: 1362-1368https://doi.org/10.1016/j.jse.2020.07.048Abstract Full Text Full Text PDF PubMed Scopus (4) Google Scholar,12Smith A.J. Bajada S. Wardle P. Morgan D. Compartment syndrome secondary to acute pectoralis major tendon rupture.J Shoulder Elbow Surg. 2015 Mar; 24: e78-81https://doi.org/10.1016/j.jse.2014.10.023Abstract Full Text Full Text PDF Scopus (4) Google Scholar, 13Thorpe S.W. Tarkin I.S. Deltoid compartment syndrome is a surgical emergency.J Shoulder Elbow Surg. 2010 Dec; 19: e11-e13https://doi.org/10.1016/j.jse.2010.05.019Abstract Full Text Full Text PDF PubMed Scopus (7) Google Scholar, 14Titolo P. Milani P. Panero B. Ciclamini D. Colzani G. Artiaco S. Acute Compartment Syndrome of the Arm after Minor Trauma in a Patient with Optimal Range of Oral Anticoagulant Therapy: A Case Report.Case Rep Orthop. 2014; 2014980940https://doi.org/10.1155/2014/980940Crossref Google Scholar Tumors are rare causes of compartment syndrome.2Barney B.M. Gillette B.P. Garces Y.I. Ding W. Cheville J.C. Rose P.S. Myeloid sarcoma of the upper extremity causing compartment syndrome: A case report.Pract Radiat Oncol. 2011 Apr-Jun; 1 (Pract Radiat Oncol): 135-138https://doi.org/10.1016/j.prro.2010.11.005Abstract Full Text Full Text PDF PubMed Scopus (4) Google Scholar,16Valone F. Liu J. Genrich G. Lattanza L. Alveolar Rhabdomyosarcoma Causing Acute Compartment Syndrome of the Forearm: A Case Report and Review of the Literature.J Hand Microsurg. 2014 Dec; 6: 92-95https://doi.org/10.1007/s12593-013-0108-0Crossref Google Scholar Because ACS of the upper extremity is an orthopedic surgical emergency, it is critical for accurate clinical identification to prevent extensive damage to tendons, muscles, and nerves.9Meshram P. Joseph J. Zhou Y. McFarland E.G. Acute compartment syndrome caused by hematoma after shoulder surgery: a case series.J Shoulder Elbow Surg. 2021 Jun; 30: 1362-1368https://doi.org/10.1016/j.jse.2020.07.048Abstract Full Text Full Text PDF PubMed Scopus (4) Google Scholar Though extremely rare, postoperative hematoma after reverse shoulder arthroplasty (RSA) can lead to compartment syndrome within 24 hours.9Meshram P. Joseph J. Zhou Y. McFarland E.G. Acute compartment syndrome caused by hematoma after shoulder surgery: a case series.J Shoulder Elbow Surg. 2021 Jun; 30: 1362-1368https://doi.org/10.1016/j.jse.2020.07.048Abstract Full Text Full Text PDF PubMed Scopus (4) Google Scholar Another risk factor for ACS is length of procedure, with a longer operative time increasing the risk for developing ACS.17Vaynrub M. Or O. Drill E. Saulan M. Boland P.J. Postoperative extremity compartment syndrome in a cancer center: Incidence and risk factors.Surg Oncol. 2021 Sep; 38101563https://doi.org/10.1016/j.suronc.2021.101563Crossref Scopus (0) Google Scholar Timely recognition and surgical intervention for compartment syndrome is critical, but diagnosis can be delayed due to lack of familiarity with uncommon presentations of compartment syndrome such as in the upper arm. In this manuscript, we describe a novel clinical situation of ACS in a 16 year-old patient four days after allograft-prosthetic composite (APC) RSA for proximal humerus osteosarcoma for the purpose of educating providers on this rare presentation of ACS of the upper extremity. An otherwise healthy 16-year-old right-hand dominant male was initially evaluated at an outside institution for indolent onset of right shoulder and arm pain. Imaging was obtained and was concerning for a pathologic lesion (Figure 1), and so he was referred to our institution. Advanced imaging and a biopsy were consistent with primary osteosarcoma of the proximal humerus (Figure 2).Figure 2MRI of the Right Humerus and Shoulder. Figure 2A. Coronal T2 weighted MRI of the R humerus demonstrating. Figure 2B. Sagittal T2 weighted MRI of the R proximal humerus again demonstrating extensive soft tissue involvement. Figure 2C. Axial T2 weighted MRI of the R proximal humeral shaft demonstrating soft involvement around the shaft of the humerus.View Large Image Figure ViewerDownload Hi-res image Download (PPT) On physical examination, he had no overlying skin changes aside from mild swelling at the biopsy site without signs of erythema. His sensation and motor function were both intact. His strength and range of motion were full but painful compared to his left shoulder. He was indicated for and subsequently underwent initial neoadjuvant chemotherapy and followed by wide surgical resection. Due to the large size of the necessary proximal humerus resection, which was to be distal to his deltoid tuberosity, APC-RSA was planned for his reconstruction. An allograft with soft tissue attachments was used to optimize postoperative function. The patient was positioned in beach chair position. The patient was then prepped and draped in usual sterile fashion and antibiotics were administered as per protocol. First the wide tumor resection was performed. A deltopectoral approach was utilized, and the biopsy site was excised. Initial dissection and exposure was focused on preserving musculotendinous attachments for eventual transfer to the allograft. The long head of the biceps, the rotator cuff tendons, the deltoid insertion, and the latissimus dorsi and teres major tendons were all tenotomized and tagged for this purpose. The tumor was noted to be intimate with the axillary and radial nerves and the brachial vessels. These were carefully preserved, and then the tumor was removed in its entirety (148cm3). Approximately 10 cm of native distal humerus remained after the resection. Intraoperative margins, including intramedullary tissue, were confirmed to be negative of any gross osteosarcoma cells, and therefore the decision was made to proceed with APC-RSA. To perform the APC-RSA (Zimmer Biomet, Warsaw, IN, USA), the glenoid was prepared and reconstructed, followed by preparation of the allograft on the back table, followed by reconstruction of the humerus with the allograft and then implantation and reduction of the final components. For the glenoid reconstruction, as there was no deformity, a non-augmented mini baseplate with a central compression screw and peripheral locking screws was used. A large, 40 mm glenosphere was impacted onto the cleaned morse taper of the baseplate; this size glenosphere was used to maximize implant stability and impingement free range of motion. For the humeral reconstruction, as previously mentioned, a humeral allograft with soft tissue attachments was utilized for the purpose of reattaching the native previously tenotomized tendons to maximize his postoperative function. This allograft was thawed and then prepared on the back table, with its length based on preoperative templating. Once prepared, it was potted with a trial stem in place onto the prepared distal humerus at 30 degrees retroversion. The allograft with the trial stem was then compressed to the native distal humerus with a 4.5 narrow locking compression plate (LCP) on the anteromedial aspect of the construct as well as a 3.5 LCP on the posterolateral aspect (DePuy Synthes, Raynham, MA, USA). After each screw was placed, the trial stem was partially removed to confirm lack of incarceration by the screw. Next, the trial stem was removed, cement inserted and pressurized, and the final humeral stem inserted. There was no cement leakage at the APC interface with pressurization, confirming excellent fit of the allograft onto the native humerus. Once the cement had hardened, multiple humeral tray and liner combinations were trialed until a final construct with optimal stability was chosen, impacted, and reduced. Finally, the patient's tenotomized tendons were reattached to the humeral allograft including the deltoid, pectoralis, latissimus and teres major, and the rotator cuff tendons. Of note, there was no active bleeding at the conclusion of the surgery. Careful hemostasis was achieved. Two deep drains were placed due to risk of hematoma formation. No transexamic acid was used in this case. The soft tissues were closed in standard fashion. Two view postoperative x-rays confirmed appropriate alignment and positioning of the components (Figure 3). The patient was admitted for standard postoperative management and monitoring. A preoperatively-placed but postoperatively-dosed supraclavicular peripheral nerve catheter (PNC) was started in the recovery room after standard postoperative neurovascular check, which was unremarkable. The highest drain output, 280 cc, occurred postoperative day (POD) two. Drain output decreased daily with the final output being 25cc serosanguinous fluid on the day of discharge. The patient was discharged home with the drains in place on postoperative POD four, with the plan to remove this in clinic at one week postoperatively. The PNC was pulled at home on POD three by the patient's parents, per standard protocol. On POD four in the early afternoon, the patient called the on call orthopedic surgery resident describing a "pop" in the shoulder during a bowel movement followed by acute worsening of pain and swelling, dressing saturation, and increased drain output over the next 30 minutes. He was instructed to present to our emergency department (ED) for evaluation. Physical examination in the ED demonstrated a saturated dressing, active serosanguinous discharge from the incision site, and numbness and weakness with a noted inability to extend his right index finger. The numbness was localized in the radial distribution, with mild decrease in the median nerve distribution as well. Sensation was intact in the axillary, lateral antebrachial, and ulnar distributions. It was also noted that the patient had a swollen, tense right arm. Stat radiographs obtained in the ED showed no signs of dislocation or hardware failure in the APC-RSA (Figure 4). Given the evolving acute compartment syndrome with the new radial nerve palsy, the patient was brought immediately to the operating room for surgical fasciotomies, hematoma evacuation, and exploration. Intracompartmental pressures were not obtained due to the clarity of the diagnosis and time sensitivity of surgical intervention. After induction of general anesthesia, the patient was again positioned in the beach chair position and standard preoperative antiseptic preparation, draping, and time-out was again performed. His previous incision was opened as was the deep fascia. There was a large hematoma which was removed from the deep compartment of the arm. The APC-RSA was intact without complication. Despite the hematoma, there were no large bleeding vessels present. All minor bleeding sites were coagulated, a new drain was placed, and the soft tissues were closed once again. On POD one, the patient was able to extend his right index finger again, and sensation in the median nerve distribution returned to normal. There continued to be mild numbness in the radial nerve distribution. On POD two, the patient reported a return to normal, full sensation in his right hand including the radial distribution, and he was discharged POD three with the drain in place. At his first follow-up appointment (POD seven), he reported minimal pain and normal sensation. The drain was removed. At his second postop-op appointment (POD 14) he continued to report little to no pain as well as intact sensation. At his third follow-up appointment (POD 21), the staples were removed, and the patient was cleared to restart chemotherapy. The patient then presented for 28-week postoperative follow-up. He had been working with physical therapy and range of motion (ROM) was 90o active forward flexion and 90 o active abduction. Passive ROM check showed 130 o forward flexion, and 130 o abduction (Figure 5A). Active external rotation with the shoulder adducted was approximately 40 o (Figure 5B). Figure 6 shows postoperative radiographs at that visit, which demonstrated a well healed APC-RSA without evidence of complication.Figure 6Postoperative radiographs at 28 weeks. There were no issues with hardware seen.View Large Image Figure ViewerDownload Hi-res image Download (PPT) In this article, we report the case of a 16 year old patient who developed upper extremity ACS secondary to hematoma formation four days after APC-RSA for proximal humerus osteosarcoma for the purpose of educating shoulder surgeons on this rare presentation of upper extremity ACS. Urgent surgical intervention, within 2 hours of the initiation of symptoms, fortunately resulted in an excellent clinical outcome. At the most recent, four-month follow-up, our patient had minimal pain, normal sensation, full motor function, and was making progress with therapy in terms of shoulder range of motion. We believe that any delay in his diagnosis or surgical intervention could have severely compromised his outcome and long-term prognosis. Surgical intervention, along with chemotherapy, has been the mainstay treatment of osteosarcoma.12Smith A.J. Bajada S. Wardle P. Morgan D. Compartment syndrome secondary to acute pectoralis major tendon rupture.J Shoulder Elbow Surg. 2015 Mar; 24: e78-81https://doi.org/10.1016/j.jse.2014.10.023Abstract Full Text Full Text PDF Scopus (4) Google Scholar However, failure of suture suspension, megaprosthesis dislocation, wound dehiscence and neurologic complications can develop postoperatively.1Angelini A. Mavrogenis A.F. Trovarelli G. Pala E. Arbelaez P. Casanova J. et al.Extra-articular shoulder resections: outcomes of 54 patients.J Shoulder Elbow Surg. 2017 Nov; 26: e337-e345https://doi.org/10.1016/j.jse.2017.04.019Abstract Full Text Full Text PDF PubMed Scopus (18) Google Scholar,18Voggenreiter G. Assenmacher S. Schmit-Neuerburg K.P. Tikhoff-Linberg procedure for bone and soft tissue tumors of the shoulder girdle.Arch Surg. 1999 Mar; 134: 252-257Crossref Scopus (38) Google Scholar Compartment syndrome is typically associated with traumatic injury but can be seen in cases of nontraumatic surgical intervention with large amounts of soft tissue damage and intraoperative blood loss.7Kim J.Y. Buck 2nd, D.W. Forte A.J. Subramanian V.S. Birman M.V. Schierle C.F. et al.Risk factors for compartment syndrome in traumatic brachial artery injuries: an institutional experience in 139 patients.J Trauma. 2009 Dec; 67: 1339-1344https://doi.org/10.1097/TA.0b013e318197b999Crossref PubMed Scopus (33) Google Scholar,10McQueen M.M. Gaston P. Court-Brown C.M. Acute compartment syndrome. Who is at risk?.J Bone Joint Surg Br. 2000 Mar; 82: 200-203Crossref PubMed Google Scholar Vaynrub et al reports that a risk factor for non-traumatic compartment syndrome in cancer patients is length of operation.17Vaynrub M. Or O. Drill E. Saulan M. Boland P.J. Postoperative extremity compartment syndrome in a cancer center: Incidence and risk factors.Surg Oncol. 2021 Sep; 38101563https://doi.org/10.1016/j.suronc.2021.101563Crossref Scopus (0) Google Scholar Resultant reperfusion can lead to acute inflammation and systemic responses from inflammatory cytokines and reactive oxygen species.5Gillani S. Cao J. Suzuki T. Hak D.J. The effect of ischemia reperfusion injury on skeletal muscle.Injury. 2012 Jun; 43: 670-675https://doi.org/10.1016/j.injury.2011.03.008Abstract Full Text Full Text PDF PubMed Scopus (155) Google Scholar In Meshram et al's case series, there were two cases of revision RTSA that led to the development of ACS, one with rheumatoid arthritis and the other osteoarthritis. The first patient developed ACS POD one secondary to a large hematoma found on emergency computed tomography scan (CT). The second patient developed multiple pulmonary emboli POD one after his revision RTSA and was started on low-molecular-weight heparin and bridge to warfarin on POD 10 and subsequently discharged home. On POD 14, the patient returned to the emergency department and due to concern for ACS underwent compartment releases from the anterior shoulder into the volar aspect of the forearm for a large hematoma. Our patient was not placed on any anticoagulation. However, he did present similarly to the second patient above, after discharge home. Providers should be aware of compartment syndrome symptoms after patient's discharge home, with a high index of suspicion. Hendy et al analyzed 6421 shoulder arthroplasties and documented that rTSA were more likely to develop postoperative hematoma, and they were more likely to develop within 21 days postoperatively.6Hendy B. Zmistowski B. Sheth M.M. Abboud J.A. Williams G.R. Namdari S. Hematoma Following Total Shoulder Arthroplasty : Incidence.Management , and Outcomes. 2017; 102: 4https://doi.org/10.22038/ABJS.2022.46679.2283Crossref Google Scholar The majority were managed nonoperatively, however, about 7% of them were associated with periprosthetic joint infections. Compartment syndrome is a potentially devastating operative complication that should be acted on immediately to prevent permanent damage to surrounding neurovascular structures. Good outcomes can be achieved with timely intervention and interdisciplinary follow-up care. This case describes an incidence of compartment syndrome following a reverse total shoulder arthroplasty and allograft prosthetic composite for treatment of osteosarcoma.