Langenskiöld Procedure for Treatment of Partial Growth Arrest of Distal Ulna After Septic Osteomyelitis: A Case Report

Abstract

Background: Although cases of impaired long bone growth due to bone and joint infections in childhood are sometimes reported, few cases of growth impairment of the ulna due to septic osteomyelitis have been described. We report herein a case of ulnar partial physeal arrest treated using the Langenskiöld procedure. Materials and Methods: A boy developed septic osteomyelitis of the right distal ulna at age 2 years 6 months. Osteomyelitis subsided after antibiotic treatment and external immobilization. As a result of impaired growth of the ulna along the long axis, shortening and trumpet-shaped deformity of the metaphysis gradually appeared. Computed tomography revealed a bony bridge, and premature epiphyseal closure due to osteomyelitis was diagnosed. The Langenskiöld procedure was performed at 4 years 4 months old. Results: As of 2 years 9 months later, no further ulnar shortening has occurred and morphological remodeling has been confirmed. Conclusions: The treatments employed for ulnar shortening include ulnar lengthening by callotasis as well as stapling of the distal radial epiphyseal line or radial shortening osteotomy. In this case, the Langenskiöld procedure proved effective because the patient was still young with growth potential and the area of the bony bridge after osteomyelitis-induced epiphyseal line damage was <30%.

Introduction

Although cases of impaired growth of long bones due to bone and joint infections in childhood are sometimes reported, few cases of growth impairment of the ulna due to septic osteomyelitis have been described. To the best of our knowledge, no previous reports have described use of the Langenskiöld procedure for premature closure of the distal ulnar epiphysis, which accounts for 20% of the growth in ulnar length.^1,2 We report herein a case of impaired growth of the distal ulna caused by septic osteomyelitis, treated with the Langenskiöld procedure.

Case Report

At age 2 years 6 months, a boy had started to complain of pain in the right wrist with no apparent cause. He subsequently developed a fever and was examined in the department of pediatrics of another hospital. The cause of the fever proved difficult to diagnose, but septic osteomyelitis of the right ulna was diagnosed based on imaging findings, and was treated using intravenous antibiotics and external immobilization with a plaster cast. The osteomyelitis subsided and wrist function appeared unaffected, but as plain radiography showed gradual progression of deformity of the distal ulna and impaired longitudinal growth, he was referred to our department at age 4 years 1 month. Initial examination revealed no pain, deformity, or functional impairment of the wrist joint.

Magnetic resonance imaging (MRI) performed when the septic osteomyelitis first developed revealed a lesion perforating the central part of the distal ulnar epiphyseal line. Plain radiography taken after the osteomyelitis had subsided showed gradually progressing deformation and shortening of the metaphysis of the distal ulna, which was approximately 5 mm shorter than the unaffected side (Figure 1). Computed tomography (CT) revealed trumpet-shaped deformity of the metaphysis and impaired longitudinal growth, with a bony bridge in the center (Figure 2). The pathophysiology was attributed to formation of a bony bridge in the center and the imposition of traction (Figure 3). The measured area of the bony bridge was <30% of the cross-sectional area of the epiphyseal line.

Figure 1.

Serial anteroposterior (AP) and lateral radiographs of the right wrist.

Note. (a) and (b) Radiographs at 1 month after septic osteomyelitis. (c) and (d) Radiographs at 3 months after septic osteomyelitis. (e) and (f) Radiographs at 1 year after septic osteomyelitis. (g) and (h) Radiographs at 1 year and 7 months after septic osteomyelitis showing gradually progressing deformation and shortening of the metaphysis of the distal ulna.

Figure 2.

(a) Computed tomography (CT) and (b) 3-dimensional CT of the wrist showing a bony bridge in the center (arrow) and trumpet-shaped deformity of the metaphysis of the ulna.

Figure 3.

A diagram illustrates the scheme of pathophysiology.

At 4 years 4 months, the Langenskiöld procedure was performed. A 1.2-mm guide pin was inserted into the ulna toward the bony bridge from the ulnar side, and drilling was conducted using 2.3- and 2.6-mm cannulated drills. As much of the bony bridge as possible was excised around the entire circumference using an otolaryngology curette under fluoroscopic guidance, and the defect was filled with subcutaneous fat (Figure 4).

Figure 4.

Operative findings: (a) Drilling was conducted toward the bony bridge from the ulnar side, and (b) the bony bridge was excised around the entire circumference using an otolaryngology curette under fluoroscopy.

Pathological results showed that the bone fragments curetted from the bony bridge contained small amounts of bone in fat and fibrous tissue. No sign of bone marrow cells was seen, and no tumorous or inflammatory lesions were present.

CT at 1 year 3 months postoperatively showed that the bony bridge had disappeared, and the surrounding healthy epiphyseal line and remnant of the epiphysis after resection of the bony bridge were also evident on MRI at 2 years 3 months postoperatively. As of 2 years 9 months after surgery, plain radiography revealed no further ulnar shortening, and morphological remodeling of the metaphysis had occurred. The distal ossific nucleus has now appeared (Figure 5). Postoperative range of motion was illustrated in Figure 6.

Figure 5.

(a) and (b) Postoperative anteroposterior (AP) and lateral radiographs, (c) and (d) AP and lateral radiographs at 9 months, (e) and (f) AP and lateral radiographs at 1 year and 3 months, and (g) and (h) AP and lateral radiographs at final 2 years 9 months follow-up showing no further ulnar shortening, and morphological remodeling of the metaphysis.

Figure 6.

Clinical photographs demonstrating (a) wrist extension, (b) wrist flexion, (c), pronation, and (d) supination at final 2 years and 9 months follow-up.

Discussions

Most reported cases of impaired growth due to premature closure of the ulnar epiphysis have been caused by either trauma, multiple cartilaginous exostoses, with a small proportion due to congenital diseases such as ulnar longitudinal deficiency or septic osteomyelitis.^3-6 Should growth impairment continue, the discrepancy between the lengths of the radius and ulna eventually results in problems such as wrist or forearm deformity and dislocation of the radial head.^7,8 After skeletal maturity, ulnar shortening is treated with ulnar lengthening by callotasis or closing wedge osteotomy of the distal radius.^4,5,7 In immature patients, carpal-adjustment surgeries such as ulnar lengthening by callotasis, stapling of the distal radial epiphyseal line, or bony bridge resection are considered.^1,4,9 To our knowledge, however, no previous reports have described the use of the Langenskiöld procedure to treat growth impairment due to premature epiphyseal closure after distal ulnar osteomyelitis.

Taking account of later remodeling, bony bridge resection is indicated if the area of the bony bridge is ≤50% of the cross-sectional area of the epiphyseal line damage and the angular deformity is ≤20°.¹⁰ In the present case, we considered the Langenskiöld procedure as likely to prove effective because the patient was still young with growth potential, the area of osteomyelitis-induced damage was <30% of the cross-sectional area of the epiphyseal line, and little angular deformity was present. However, secondary surgery may be required if cosmetic deformity arises due to ulnar shortening, or pain and a functional deficit occur. Under such conditions, the patient must be monitored until skeletal maturity.