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Long-Term Follow-up After Limb Salvage in Skeletally Immature Children with a Primary Malignant Tumor of the Distal End of the Femur

¹ Department of Orthopaedic Surgery, Hyogo College of Medicine, 1-1 Mukogawa Nishinomiya, Hyogo 663-8501, Japan. E-mail address: h-futani{at}hyo-med.ac.jp
² Department of Orthopedic Surgery, Tottori University, 36-1 Nishimachi Yonago, Tottori 683-8504, Japan
³ Department of Orthopaedic Surgery, Gifu University, 40 Tsukasa-machi, Gifu, Gifu 500-8705, Japan
⁴ Department of Orthopaedic Surgery, Teikyo University School of Medicine, 2-11-2 Kaga, Itabashi-ku, Tokyo 173-8605, Japan
⁵ Department of Orthopaedic Surgery, Keio University School of Medicine, 35 Shinano-machi, Shinjuku-ku, Tokyo 160-8582, Japan
⁶ Department of Orthopaedic Surgery, Osaka University, 2-2 Yasuda-oka, Suita, Osaka 565-0871, Japan

Investigation performed at the Department of Orthopaedic Surgery, Hyogo College of Medicine, Hyogo, Japan

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NOTE: The authors thank their colleagues in the Japanese Musculoskeletal Oncology Group for their participation in the present study: H. Yoshida, MD, Nihon University; K. Yonemura, MD, Kumamoto University; K. Isu, MD, National Sapporo Hospital; K. Yokoyama, MD, National Kokura Hospital; N. Araki, MD, Osaka University; T. Goto, MD, The University of Tokyo; A. Kawai, MD, National Cancer Center Hospital; T. Ozaki, MD, Okayama University; S. Tatezaki, MD, Chiba Cancer Center Hospital; T. Hotta, MD, Niigata University; T. Tajino, MD, Fukushima Medical University; S. Nasuno, MD, Kitasato University; E. Chousa, MD, Miyazaki Medical College; H. Kakizaki, MD, National Hirosaki Hospital; K. Shinjo, MD, Nagoya National Hospital; N. Ueda, MD, Osaka Medical College; Y.H. Lee, MD, Tokai University; H. Watanabe, MD, Gunma University; H. Murata, MD, Kyoto Prefectural University of Medicine; K. Ihara, MD, Yamaguchi University; T. Nakagawa, MD, Kansai Medical University; H. Isaki, MD, National Defense Medical College; T. Nishikori, MD, Shimane Medical University; T. Takagi, MD, Juntendo University; A. Imakiire, MD, Tokyo Medical University; H. Tsuchiya, MD, Kanazawa University; Y. Aoki, MD, Osaka National Hospital; T. Nagai, MD, Saitama Medical School; H. Kuroda, MD, Tokyo Medical and Dental University; and M. Hatori, MD, Tohoku University.

In support of their research for or preparation of this manuscript, one of the authors (H.F.) received grants or outside funding from the Fund for Cancer Research from Hyogo Prefecture Health Promotion Association. None of the authors received payments or other benefits or a commitment or agreement to provide such benefits from a commercial entity. No commercial entity paid or directed, or agreed to pay or direct, any benefits to any research fund, foundation, educational institution, or other charitable or nonprofit organization with which the authors are affiliated or associated.

Methods: The cases of forty children were retrospectively reviewed in a multicenter study based on the responses to a questionnaire. Twenty-eight patients had had endoprosthetic reconstruction, and twelve had had biological reconstruction. Functional evaluation was based on the Musculoskeletal Tumor Society scoring system, with numerical values from 0 to 5 points assigned for each of the following six categories: pain, function, emotional acceptance, use of supports, walking ability, and gait. These values were added, and the functional score was presented as a percentage of the maximum possible score. Limb-length discrepancy was measured with orthoroentgenograms. Complications and their treatment were analyzed. Patient survival and the survival of the reconstructions were analyzed with use of the Kaplan-Meier method.

Results: Seven patients died and thirty-three remained alive, for a survival rate of 82% at ten years postoperatively. For the surviving patients, the mean follow-up periods (and standard deviations) were similar for the twenty-two who had endoprosthetic reconstruction (13.2 ± 3.9 years) and the eleven who had biological reconstruction (10.4 ± 4.4 years). All patients had reached skeletal maturity. The mean final functional score was 74% ± 18% in the endoprosthetic reconstruction group and 68% ± 17% in the biological reconstruction group (p = 0.37). For the nineteen patients who underwent limb-lengthening, the mean functional score increased significantly from 65% ± 21% before the procedure to 81% ± 11% after the lengthening (p = 0.0016). There were five early and twenty-eight late complications. In the endoprosthetic reconstruction group, the most frequent complications were deep infection and aseptic loosening. In the biological reconstruction group, the most frequent complications were implant breakage and nonunion. Revision surgeries were required in seventeen patients, including five who had an amputation. The rate of survival of the endoprosthetic reconstructions was 77% at five years and 51% at ten years postoperatively, whereas the rate of survival of the biological reconstructions was 46% at both five and ten years postoperatively.

Conclusions: Endoprosthetic or biological reconstructions as limb salvage provided good functional outcome in skeletally immature children with a malignant bone tumor of the distal aspect of the femur despite a high rate of revisions and limb-lengthening procedures.

Level of Evidence: Therapeutic Level III. See Instructions to Authors for a complete description of levels of evidence.

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