Abstract
Incidence of chronic osteomyelitis in the humerus is relatively rare compared with the incidence in the lower limbs, with the humerus being the most commonly affected bone in the upper extremity.2Birt M.C. Anderson D.W. Bruce Toby E. Wang J. Osteomyelitis: recent advances in pathophysiology and therapeutic strategies.J Orthop. 2016; 14: 45-52https://doi.org/10.1016/j.jor.2016.10.004Crossref PubMed Scopus (93) Google Scholar,31Street M. Crawford H. Pediatric humeral osteomyelitis.J Pediatr Orthop. 2015; 35: 628-633https://doi.org/10.1097/BPO.0000000000000347Crossref PubMed Scopus (7) Google Scholar,35Wang X. Yu S. Sun D. Fu J. Wang S. Huang K. et al.Current data on extremities chronic osteomyelitis in southwest China: epidemiology, microbiology and therapeutic consequences.Sci Rep. 2017; 7: 16251https://doi.org/10.1038/s41598-017-16337-xCrossref PubMed Scopus (17) Google Scholar Management of humeral osteomyelitis includes bone debridement, delayed defect reconstruction, and suppressive antibiotic therapy.10Haidar R. Der Boghossian A. Atiyeh B. Duration of post-surgical antibiotics in chronic osteomyelitis: empiric or evidence-based?.Int J Infect Dis. 2010; 14: e752-e758https://doi.org/10.1016/j.ijid.2010.01.005Abstract Full Text Full Text PDF PubMed Scopus (68) Google Scholar The chronic course of infections and repeated surgeries often lead to severe soft-tissue compromise, neurologic deficit (radial nerve), and poor functional results.4Chhabra A.B. Golish S.R. Pannunzio M.E. Butler Jr., T.E. Bolano L.E. Pederson W.C. Treatment of chronic nonunions of the humerus with free vascularized fibula transfer: a report of thirteen cases.J Reconstr Microsurg. 2009; 25: 117-124https://doi.org/10.1055/s-0028-1090624Crossref PubMed Scopus (15) Google Scholar,35Wang X. Yu S. Sun D. Fu J. Wang S. Huang K. et al.Current data on extremities chronic osteomyelitis in southwest China: epidemiology, microbiology and therapeutic consequences.Sci Rep. 2017; 7: 16251https://doi.org/10.1038/s41598-017-16337-xCrossref PubMed Scopus (17) Google Scholar The definitive treatment for chronic humerus osteomyelitis often includes removal of the compromised bone segment. Treatment of bone defects is a surgical challenge for orthopedic surgeons. Among the most used surgical alternatives are free vascularized fibula transfer, callus distraction with circular external fixation or rail fixator, and induced membrane technique (IMT).7Ferreira N. Marais L.C. Serfontein C. Two stage reconstruction of septic non-union of the humerus with the use of circular external fixation.Injury. 2016; 47: 1713-1718https://doi.org/10.1016/j.injury.2016.06.014Abstract Full Text Full Text PDF PubMed Scopus (9) Google Scholar,13Lakhani A. Singh D. Singh R. Outcome of rail fixator system in reconstructing bone gap.Indian J Orthop. 2014; 48: 612-616https://doi.org/10.4103/0019-5413.144237Crossref PubMed Scopus (8) Google Scholar,17Liu T. Zhang X. Li Z. Zeng W. Peng D. Sun C. Callus distraction for humeral nonunion with bone loss and limb shortening caused by chronic osteomyelitis.J Bone Joint Surg Br. 2008; 90: 795-800https://doi.org/10.1302/0301-620X.90B6.20392Crossref PubMed Scopus (29) Google Scholar,27O'Connor C.M. Perloff E. Drinane J. Cole K. Marinello P.G. An analysis of complications and bone defect length with the use of induced membrane technique in the upper limb: a systematic review.Hand (N Y). 2020; 15 (1558944720918368. https://doi.org/10.1177/1558944720918368)Google Scholar The IMT, otherwise known as the Masquelet technique, is a useful procedure27O'Connor C.M. Perloff E. Drinane J. Cole K. Marinello P.G. An analysis of complications and bone defect length with the use of induced membrane technique in the upper limb: a systematic review.Hand (N Y). 2020; 15 (1558944720918368. https://doi.org/10.1177/1558944720918368)Google Scholar for bone regeneration in the metaphyseal and diaphyseal regions of long bones.8Gouron R. Deroussen F. Juvet-Segarra M. Plancq M.C. Collet L.M. Reconstruction of congenital pseudarthrosis of the clavicle with use of the Masquelet technique: a case report.JBJS Case Connect. 2012; 2: e77https://doi.org/10.2106/JBJS.CC.L.00095Crossref PubMed Google Scholar Since 2000, Masquelet et al19Masquelet A.C. Induced membrane technique: pearls and pitfalls.J Orthop Trauma. 2017; 31: S36-S38https://doi.org/10.1097/BOT.0000000000000979Crossref PubMed Scopus (70) Google Scholar, 20Masquelet A.C. Begue T. The concept of induced membrane for reconstruction of long bone defects.Orthop Clin North Am. 2010; 41: 27-37https://doi.org/10.1016/j.ocl.2009.07.011Abstract Full Text Full Text PDF PubMed Scopus (415) Google Scholar, 21Masquelet A.C. Fitoussi F. Begue T. Muller G.P. Reconstruction of the long bones by the induced membrane and spongy autograft.Ann Chir Plast Esthet. 2000; 45: 346-353PubMed Google Scholar have used this technique for segmental bone loss, bone tumors, and septic nonunions. IMT allows the repair of large segmental bone defects without the application of distraction osteogenesis or microvascular surgery.16Litvina E.A. Semenistyy A.A. A case report of extensive segmental defect of the humerus treated with Masquelet technique.J Shoulder Elbow Surg. 2020; 29: 1368-1374https://doi.org/10.1016/j.jse.2020.03.018Abstract Full Text Full Text PDF PubMed Scopus (1) Google Scholar The IMT involves two stages. In the first stage, a polymethyl methacrylate spacer (with or without antibiotics) is placed into the bone defect to induce the formation of a vascularized membrane. The second stage is performed 6 weeks later and consists of filling in the gap with a bone graft.9Haddad B. Zribi S. Haraux E. Deroussen F. Gouron R. Klein C. Induced membrane technique for clavicle reconstruction in paediatric patients: Report of four cases.Orthop Traumatol Surg Res. 2019; 105: 733-737https://doi.org/10.1016/j.otsr.2019.03.010Abstract Full Text Full Text PDF PubMed Scopus (3) Google Scholar This technique replaces almost any long bone defect in a fixed amount of time.16Litvina E.A. Semenistyy A.A. A case report of extensive segmental defect of the humerus treated with Masquelet technique.J Shoulder Elbow Surg. 2020; 29: 1368-1374https://doi.org/10.1016/j.jse.2020.03.018Abstract Full Text Full Text PDF PubMed Scopus (1) Google Scholar This article presents a clinical case report of a patient treated with IMT augmented with fibula autograft for an 18-cm humerus shaft defect due to a pathologic fracture nonunion on chronic osteomyelitis. The patient provided informed consent. The patient was a 39-year-old woman, a healthy adult patient, right-handed, who worked as a cook. In January 2016, she fell off a bicycle with a direct blow to the shoulder and right arm, for which she went to an emergency room where she was diagnosed with shoulder contusion (Fig. 1). In February 2016 (25 days later), she was rushed to the emergency room for persistent pain that was not responding to oral analgesics. Previously, she had been evaluated by a general practitioner, including radiography (informed as normal) and ultrasound that revealed a subacromial and subdeltoid bursitis and long head biceps tendinitis (Fig. 2). Upon physical examination, there was severe pain in the lateral aspect of the arm, with loss of active abduction, preserved active rotations, and painless passive shoulder rotations, without edema or erythema. Inflammatory parameters showed leukocytosis (16.4 × 103/μL; normal range: 4.5–11.5 × 103/μL), increased erythrocyte sedimentation rate (ESR) (116 mm/h; normally less than 20 mm/h), and increased C-reactive protein (CRP) (124 mg/L; normally less than 5 mg/L). Analgesia was optimized, and consultation was indicated in case of increased volume, erythema, fever, local heat, or increased pain. In March 2016, she consulted for a sudden increase in pain and functional impotence without trauma. The radiography showed a proximal diaphyseal humerus fracture on the pathological bone with cortical disaggregation through the entire humerus, highly indicative of malignancy (Fig. 3). A shoulder immobilizer was placed, a humerus computed tomography scan was requested, and an evaluation by the orthopedic oncology unit was programed. This unit also requested a computed tomography scan of the chest, abdomen, pelvis, and bone scintigraphy (technetium-99m). The studies ruled out metastatic disease, with radioisotope hypercaptation at the level of the right humerus, so it was decided to perform a biopsy. Nevertheless, after 5 days, she consulted again because of a large increase in volume and purulent discharge from the medial right arm. The patient underwent a surgical cleaning through an anterolateral approach to the humerus, with debridement of necrotic tissue and abundant irrigation with physiological serum. The bone was biopsied, reported as chronic suppurative osteomyelitis with abscess, negative for neoplasia at the cytological examination, and negative for aerobic and anaerobic cultures. Endovenous antibiotics treatment was performed with cefazolin 1 g tid for 2 weeks, with a significant decrease in leukocytes (8.2 × 103/μL), ESR (62 mm/h), and CRP (16 mg/L). Cloxacillin 1 g qid orally was prescribed, and a functional brace was installed to manage the fracture. Finally, the patient was discharged. The functional brace was poorly tolerated because of the persistence of purulent discharge, with the patient’s low adherence to the oral antibiotic treatment and without attending medical checkups. In June 2016, the patient was admitted to the emergency room because of an increase in volume (abscess), abundant purulent discharge, and altered inflammatory parameters (leukocytes 13.8 × 103/μL; ESR 50 mm/h.; CPR 122 mg/L) (Fig. 4). She was hospitalized for a surgical cleaning; microbiological samples were taken, and intravenous antibiotic was administered (cefazolin 2 g tid and gentamicin 160 mg qd for 11 days). Bone cultures were negative, and inflammatory parameters had decreased (leukocytes, 8.5 × 103/μL; ESR, 30 mm/h; CPR, 6 mg/L). The patient was discharged again with long-term suppressive oral antibiotics (flucloxacillin 500 mg tid) and a functional brace. In serial radiography, a progressive area of compromise is evidenced, evolving to panhumerus osteomyelitis with the progression of bone loss (Fig. 5). In February 2017, the patient was admitted to the emergency room because of increased volume (abscess) and a new draining fistula with purulent discharge. She was hospitalized again for a surgical cleaning; cultures were taken, and an intravenous antibiotic was administered (cefazolin 2 g tid and gentamicin 160 mg qd for 21 days). The patient was discharged once again with long-term suppressive oral antibiotics (flucloxacillin 500 mg tid). After 12 months of irregular medical checkups and long-term suppressive antibiotic therapies with irregular adherence, an evaluation by the shoulder and elbow unit was requested because of persistent purulent drainage through two draining fistulas. The diagnosis was humeral shaft nonunion, extensive humeral bone defect, and chronic osteomyelitis Cierny-Mader IV secondary to nonaggressive management of the infection (Fig. 6). In addition, an magnetic resonance imaging (MRI) study of the humerus showed complete diaphyseal involvement without compromise of the epiphysis (Fig. 7). The preoperative inflammatory parameters were leukocytes, 11.1 × 103/μL; ESR, 25 mm/h; and CPR, 22 mg/L. Complete humeral resection of the devitalized and infected tissue was performed to decide definitively on reconstructive management. After surgical irrigation and debridement, all devitalized fragments were removed, and a proximal and distal bone resection, conserving the minimal humeral shaft bone stock for osteosynthesis, was performed (The minimal osteosynthesis was planned with an LCP Extra-articular Distal Humerus Plate [DePuy Synthes, Oberdorf, Switzerland]), considering a proximal molding to achieve the placement of two locked screws to the humeral head. At least one bicortical screw was considered for the diaphysis in the distal humerus, fixing the distal locked screws to the lateral column. The removed bone was biopsied and cultivated. The proximal and distal medullary canals were also cultivated. The biopsy showed nonspecific chronic osteomyelitis, with persistent acute activity without granulomas or tumor, on both ends. One bone culture of the distal humerus showed multisensitive coagulase-negative Staphylococcus. Other cultures (two distal and three proximal) were negative. Medullary canal cultures were also negative. After marginal resection of the main fragments, the bone defect was 18 cm (Fig. 8). Among the treatment alternatives, the following were evaluated:•Callus distraction osteogenesis with a circular external fixator (Ilizarov). Both proximal and distal fixation are scarce, and to perform the osteotomy in both foci, the fragment to be transported is exceedingly small. Thus, the reconstruction and bone transport unit considered this case not suitable for bone transport, with a high probability of failure due to scarce bone fixation.•Free vascularized fibula transfer was a feasible alternative for this case, but fixation is scarce, and the team’s experience with this technique was limited.•The IMT was considered as an excellent alternative, but fixation with the plate alone would likely have been precarious.•Nonvascularized fibula autograft was considered as another appropriate alternative. The team had vast experience, with excellent results, using autograft/allograft of the fibula. The use of a standardized technique reduces donor site morbidity, lowers costs, increases graft incorporation, and reduces other types of complications associated with allografts (eg, infection). Finally, it was decided to perform a nonvascularized fibula autograft to augment the fixation of the segment (using the mechanical properties of the fibula and a plate) and maintain an adequate environment for the IMT (biological stimulation and new bone). Although the osteotomy did not show osteomyelitis-free margins, the maximum possible bone resection was considered adequate fixation for the final construct using the MRI study. In case of residual infection foci, the use of long-term suppressive antibiotic treatment on a better irrigated epiphyseal bone was also considered. The intravenous antibiotic was administered (cefazolin 1 g tid), and a second surgical cleaning was performed at 72 hours. A polymethylmethacrylate antibiotic-loaded cement spacer was left (Simplex P with 1 g Tobramycin Stryker, Mahwah, NJ). Bone cultures were taken (three for each end), which were negative. After 14 days of intravenous antibiotics from admission, a third surgical cleaning was performed without suppurative wounds by placing a cement spacer with an endomedullary fixation (first reconstructive stage), thus completing 21 days of antibiotics by indication of the infectious disease service. The patient was finally discharged with downward inflammatory parameters (leukocytes 9.1 × 103/μL, ESR 61 mm/h, CRP 22 mg/L). Cement spacer removal, a surgical cleaning involving the removal of necrotic tissue and irrigation, with 6-liter physiological saline and a pulsatile lavage system, was performed. After performing hemostasis, a cement spacer (Surgical Simplex P Stryker) of equal diameter as the diaphyseal humerus was installed with a central axis of a Steinmann 3.0 pin (Fig. 9). All intraoperative bone cultures were negative (three for each end). The patient was advised to attend the second stage in 6 weeks. Instead, she resumed follow-up 5 months after the first stage. Preoperative inflammatory parameters were leukocytes, 11.3 103/μL; ESR, 15 mm/h; and CPR, 6 mg/L. The patient was placed in a prone position, with a wide posterior humerus approach. Paratricipital dissection, identification, and protection of the radial and axillary nerves during surgery, all wrapped in scar tissue, were carried out. Cement spacer was removed, respecting the fibrous membrane envelope, and preserving the zone of the radial nerve (Fig. 10A). Proximal and distal humerus ends were checked without secretion. The necrotic bone was resected, and the remaining bone was curetted of bleeding (Paprika sign). Simultaneously, in a lateral leg approach, respecting superficial peroneal nerve and syndesmal ligament complex, proximal and distal fibula osteotomies were performed. They were harvested for autograft of length 20 cm, preserving the periosteum intact. Fibula autograft was positioned, checking adequate length according to surgical planning (Fig. 10B). The ends of the fibula were carved to achieve an endomedullary position at each end. The previously planned 10-hole plate (SYNTHES LCP Extra-articular Distal Humerus Plate, Oberdorf, Switzerland) was first molded at its proximal end and then placed inside the membrane. Three compression cortical screws were fixed into the fibula to compress it against the cortices of the humerus extremes (Fig. 11A). Then, definitive fixation was obtained with two locked screws proximal to the humeral head, with one cortical and four locked to the lateral column on the distal humerus, achieving adequate stability. Then, the membranous cavity was filled with a morcellated cancellous bone allograft from four femoral heads, mixed with 4 g of vancomycin (Figure 11, Figure 12A). The fibrovascular membrane was closed with self-locking running stitches of 3.0 Vicryl suture (Fig. 12B). Finally, closure by planes was performed. Postoperatively, the patient evolved with radial and axillary nerve paresis. All six intraoperative cultures were negative (two proximal bone humerus, one proximal medullary canal, two distal bone humerus, and one distal medullary canal). Postoperative radiographs showed adequate fixation (Fig. 13). The patient had an adequate perioperative evolution, so she was discharged after a week and given cefazolin 1 g tid and ciprofloxacin 500 mg bid. Upon discharge, she received oral antibiotic treatment of cefadroxil 1 g bid. The arm was immobilized with a sling and a dynamic extension orthosis for radial nerve neuropraxia. Control at 10 days showed no complications of operative wounds or signs of infection, with tolerable pain. Progressive rehabilitation was started by both a physiotherapist and occupational therapist. After 2 months, she had partial improvement in the function of the radial and axillary nerves. The use of cefadroxil 1 g bid was maintained for 12 months. Graft consolidation was noted on the postoperative radiograph after 12 weeks. On the follow-up visit after 2 years, complete consolidation of the fibular autograft and morcellated cancellous bone allograft was observed (Fig. 14) without motor or sensory deficit in the radial territory and only mild hypoesthesia in the axillary territory. Physical examination showed an adequate range of motion of the shoulder (170° anterior elevation; 130° abduction; 50° external rotation; internal rotation L2) (Fig. 15), elbow with normal range of motion, with isolated 15° loss of extension, and a Disabilities of the Arm, Shoulder and Hand Score (DASH Score) of 8.3. She will continue with annual checkups, or emergency consultations, in case of reactivation of the infection. Acute osteomyelitis of the humerus in adult patients is a diagnostic challenge. Suspicion and early diagnosis are key to adequate medical-surgical treatment to avoid associated complications, such as chronic osteomyelitis. Furthermore, when an infected nonunion and a large humeral defect are added, treatment alternatives become limited. Acute osteomyelitis in adults is rare and generally linked to local and general comorbidities (trauma, bone surgery, joint replacement, immunodeficiency, diabetes, or vascular disease).15Lew D.P. Waldvogel F.A. Osteomyelitis.Lancet. 2004; 364: 369-379https://doi.org/10.1016/S0140-6736(04)16727-5Abstract Full Text Full Text PDF PubMed Scopus (1184) Google Scholar A hematogenous spread of bacteria leading to osteomyelitis is exceptional in adult patients but more frequent in the elderly.15Lew D.P. Waldvogel F.A. Osteomyelitis.Lancet. 2004; 364: 369-379https://doi.org/10.1016/S0140-6736(04)16727-5Abstract Full Text Full Text PDF PubMed Scopus (1184) Google Scholar The reported case presented blunt trauma to the arm, so a hyperemic area was probably generated with the consequent hematogenous bacterial spread. Staphylococcus aureus and by the most associated with bone osteomyelitis because of to which are to with M. The of infections associated to orthopedic 2014; 31: PubMed Google J. C. J. J. J. et osteomyelitis in 31: PubMed Scopus Google Scholar Other are not frequent and for only H. of new on reported PubMed Scopus Google Scholar of the is In the reported case, out of all the bone cultures taken, only one was for coagulase-negative which have been a This it to antibiotic probably associated with the and use of antibiotics cultures in this to recent treatment the of hematogenous osteomyelitis for weeks and be by oral for B. E. management of chronic J Surg. 2004; Full Text Full Text PDF PubMed Scopus Google C. K. C. W. H. 2019; Scopus Google Scholar a approach. are at the of infection by debridement and appropriate antibiotic D.P. Waldvogel F.A. Osteomyelitis.Lancet. 2004; 364: 369-379https://doi.org/10.1016/S0140-6736(04)16727-5Abstract Full Text Full Text PDF PubMed Scopus (1184) Google B. E. management of chronic J Surg. 2004; Full Text Full Text PDF PubMed Scopus Google C. K. C. W. H. 2019; Scopus Google Scholar In this case, the infectious disease decided on a after humeral resection, considering that the patient adherence and surgical of the infection. a of general fever, and local and bone on are signs of a local infectious D.P. Waldvogel F.A. Osteomyelitis.Lancet. 2004; 364: 369-379https://doi.org/10.1016/S0140-6736(04)16727-5Abstract Full Text Full Text PDF PubMed Scopus (1184) Google Scholar Although patient did not during entire clinical second emergency she presented functional and with inflammatory the when compared with the first consultation showed with osteomyelitis. In this the management had to be with debridement and cleaning of the medullary cavity and intravenous antibiotic probably have been at this Nevertheless, hematogenous osteomyelitis and or metastatic bone by imaging be in long Bone Joint Surg Am. 2004; PubMed Scopus Google S. T. T. T. H. M. diagnosis osteomyelitis and bone 2008; PubMed Scopus Google Scholar The in osteomyelitis are of tissue, or which are also observed in bone M. H. M. biopsy of bone and tissue for surgical Orthop 2012; PubMed Scopus (9) Google S. T. T. T. H. M. diagnosis osteomyelitis and bone 2008; PubMed Scopus Google Scholar by MRI would likely infectious and but it not provided for osteomyelitis. the of a or metastatic bone had to be by M. H. M. biopsy of bone and tissue for surgical Orthop 2012; PubMed Scopus (9) Google S. T. T. T. H. M. diagnosis osteomyelitis and bone 2008; PubMed Scopus Google Scholar bone defects are an for orthopedic J. R. J. Treatment of bone loss with the induced membrane technique: and Orthop Trauma. 2015; 29: PubMed Scopus Google Scholar treatment is not and there are for its segment membrane technique, and vascularized fibula E. S. Masquelet technique: or A systematic and 2016; 47: Full Text Full Text PDF PubMed Scopus Google Scholar The membrane technique, by is a surgical technique used to manage diaphyseal bone defects of two stages. In the first stage, a debridement and resection of necrotic bone tissue are performed to a bleeding bone known as the with the use of a polymethylmethacrylate spacer to the bone defect, which is with an external fixator or another osteosynthesis The spacer allows the filling of the bone defect and the of a membrane in to a by the This fibrovascular which been in is in vascular and N. Zhang X. Wang et induced membrane in a femoral defect in a Rep. PubMed Scopus Google Masquelet A.C. R. J. Induced including vascular and and bone Orthop Res. 2004; PubMed Scopus Google Z. M. E. D. S. Masquelet technique: of spacer and on and bone 2019; 47: PubMed Scopus (17) Google Scholar In a second stage, 6 to weeks an is on the the spacer is removed, and the bone defect is filled with an morcellated Finally, is to the construct with an endomedullary or This technique a rate of most of on the and and on the upper E. S. Masquelet technique: or A systematic and 2016; 47: