This study aimed to characterize affected-limb pain and its associated factors among adolescent and young adult (AYA) survivors of lower extremity osteosarcoma in Japan. We hypothesized that both physical and psychosocial factors would be associated with pain, with an interaction between pain intensity and pain interference.
Methods:
A cross-sectional study was conducted among osteosarcoma survivors aged 18–39 years who had completed treatment at least 2 years earlier. Self-administered questionnaires assessed pain intensity and interference, physical function, fatigue, sleep disturbance, anxiety, depression, and perceived social support. Multiple regression analyses were performed to identify factors associated with pain outcomes. Semistructured interviews were also conducted to provide supplementary insights into pain characteristics and contextualize the quantitative findings.
Results:
Among 64 participants, 65.6% reported pain or pain interference during the preceding 7 days. Pain intensity was significantly associated with poorer physical function, older age at diagnosis, and lower perceived social support, whereas pain interference was primarily associated with greater fatigue severity. Pain intensity and pain interference were strongly correlated. Qualitative analysis identified four pain characteristics prosthesis-related pain, phantom limb pain, secondary pain, and weather-related pain. Interviews further illustrated how physical limitations, fatigue, and social support shaped pain experiences.
Conclusion:
Pain among long-term osteosarcoma survivors is influenced by multiple biopsychosocial factors. Although average pain severity was relatively mild, substantial interindividual variability was observed. Comprehensive survivorship care should address physical function, fatigue management, and social support to optimize long-term pain management and self-care among AYA osteosarcoma survivors.
AnghelescuDL, SteenBD, WuH, et al.Prospective study of neuropathic pain after definitive surgery for extremity osteosarcoma in a pediatric population. Pediatr Blood Cancer2017;64(3); doi: 10.1002/pbc.26162
2.
AllenJM, NielK, GuoA, et al.Psychosocial factors and psychological interventions: Implications for chronic post-surgical pain in pediatric patients with osteosarcoma. J Clin Psychol Med Settings2021;28(3):468–476; doi: 10.1007/s10880-020-09748-y
3.
FurtadoS, GrimerRJ, CoolP, et al.Physical functioning, pain and quality of life after amputation for musculoskeletal tumours: A national survey. Bone Joint J20152015;97-B(9):1284–1290; doi: 10.1302/0301-620X.97B9.35192
4.
KatsumotoS, MaruM, YonemotoT, et al.Uncertainty in young adult survivors of childhood and adolescent cancer with lower-extremity bone tumors in Japan. J Adolesc Young Adult Oncol2019;8(3):291–296; doi: 10.1089/jayao.2018.0120
5.
TreedeRD, RiefW, BarkeA, et al.A classification of chronic pain for ICD-11. Pain2015;156(6):1003–1007; doi: 10.1097/j.pain.0000000000000160
6.
TaylorMF, PooleyJA. Sarcoma survivors’ perspectives on their body image and functional quality of life post-resection/limb salvage surgery. Eur J Cancer Care (Engl)2017;26(6):e12667; doi: 10.1111/ecc.12667
7.
OguraK, KawanoH, ShimoseS, et al.Osteosarcoma in Japan: Report from the bone and soft tissue tumor registry 2006–2022. Jpn J Clin Oncol2025;55(9):1046–1053; doi: 10.1093/jjco/hyaf094
8.
Foundation for Promotion of Cancer Research. Cancer statistics in Japan 2024. Tokyo; 2024.
9.
YamamotoN, ArakiY, TsuchiyaH. Joint-preservation surgery for bone sarcoma in adolescents and young adults. Int J Clin Oncol2023;28(1):12–27; doi: 10.1007/s10147-022-02154-4
10.
HanG, BiWZ, XuM, et al.Amputation versus limb-salvage surgery in patients with osteosarcoma: A meta-analysis. World J Surg2016;40(8):2016–2027; doi: 10.1007/s00268-016-3500-7
11.
HussonO, ZebrackBJ. Psychometric evaluation of an adolescent and young adult module of the impact of cancer instrument. J Adolesc Young Adult Oncol2017;6(1):159–170; doi: 10.1089/jayao.2016.0038
12.
FauskeL, BondevikH, BrulandØS, et al.Negative and positive consequences of cancer treatment experienced by long-term osteosarcoma survivors: A qualitative study. Anticancer Res2015;35(11):6081–6090.
13.
HeinemannM, HoffmannC, HardesJ, et al.Pain in survivors of Ewing sarcoma: Prevalence, associated factors and prediction of recurrence. Pediatr Blood Cancer2021;68(3):e28801; doi: 10.1002/pbc.28801
14.
KumarB, SharmaP, ShantanuK, et al.Limb salvage strategy amendment for a better future in the era of bone cancer therapy: A cross-sectional study in North India. Cureus2023;15(7):e41768; doi: 10.7759/cureus.41768
15.
KufflerDP. Coping with phantom limb pain. Mol Neurobiol2018;55(1):70–84; doi: 10.1007/s12035-017-0718-9
16.
WongSS, SvingosAM, HeatonSC. Behavioral therapy for acute post-surgical phantom limb pain in a young adult with osteosarcoma. Pain Pract2019;19(5):571–572; doi: 10.1111/papr.12768
17.
DeMossP, RamseyLH, KarlsonCW. Phantom limb pain in pediatric oncology. Front Neurol2018;9:219; doi: 10.3389/fneur.2018.00219
18.
RajaS, CarrD, CohenM, et al.The revised International Association for the Study of Pain definition of pain: Concepts, challenges, and compromises. Pain2020;161(9):1976–1982; doi: 10.1097/j.pain.0000000000001939
19.
AlbertsNM, LeisenringW, WhittonJ, et al.Characterization of chronic pain, pain interference, and daily pain experiences in adult survivors of childhood cancer: A report from the Childhood Cancer Survivor Study. Pain2024;165(11):2530–2543; doi: 10.1097/j.pain.0000000000003284
20.
BeversK, WattsL, KishinoND, et al.The biopsychosocial model of the assessment, prevention, and treatment of chronic pain. US Neurol2016;12:98–104; doi: 10.17925/USN.2016.12.02.98
21.
KarlsonCW, AlbertsNM, LiuW, et al.Longitudinal pain and pain interference in long-term survivors of childhood cancer: A report from the Childhood Cancer Survivor Study. Cancer2020;126(12):2915–2923; doi: 10.1002/cncr.32853
22.
WilsonM. Integrating the concept of pain interference into pain management. Pain Manag Nurs2014;15(2):499–505; doi: 10.1016/j.pmn.2011.06.004
23.
EdelenMO, RodriguezA, HermanPM, et al.Crosswalking the patient-reported outcomes measurement information system physical function, pain interference, and pain intensity scores to the Roland-Morris disability questionnaire and the Oswestry disability index. Arch Phys Med Rehabil2021;102(7):1317–1323; doi: 10.1016/j.apmr.2021.02.014
24.
CellaD, ChoiS, GarciaS, et al.Setting standards for severity of common symptoms in oncology using the PROMIS item banks and expert judgment. Qual Life Res2014;23(10):2651–2661; doi: 10.1007/s11136-014-0732-6
DavisAM, WrightJG, WilliamsJI, et al.Development of a measure of physical function for patients with bone and soft tissue sarcoma. Qual Life Res1996;5(5):508–516; doi: 10.1007/BF00540024
27.
OguraK, UeharaK, AkiyamaT, et al.Cross-cultural adaptation and validation of the Japanese version of the Toronto Extremity Salvage Score (TESS) for patients with malignant musculoskeletal tumors in the lower extremities. J Orthop Sci2015;20(6):1098–1105; doi: 10.1007/s00776-015-0767-8
28.
HattaH, HigashiA, YashiroH, et al.A validation of the hospital anxiety and depression scale [in Japanese]. Jpn J Psycho Som Med1998;38:309–315; doi: 10.15064/jjpm.38.5_309
29.
IwasaH, GondoK, MasuiY, et al.Reliability and validity of the Japanese version of the Social Support Scale—Study targeting middle-aged and older adults -[in Japanese]. J Health Welf Stat2007;54(6):26–33.
30.
BraunV, ClarkeV. Thematic analysis: A practical guide. Sage: London; 2022.
31.
PattonM, ForsterVJ, ForbesC, et al.Characterizing pain in long-term survivors of childhood cancer. Support Care Cancer2022;30(1):295–303; doi: 10.1007/s00520-021-06386-4
32.
HendersonER, GroundlandJS, PalaE, et al.Failure mode classification for tumor endoprostheses: Retrospective review of five institutions and a literature review. J Bone Joint Surg Am2011;93(5):418–429; doi: 10.2106/JBJS.J.00834
33.
RamosO, MierkeA, FillerR, et al.Outpatient opioid prescribing habits in pediatric patients with bone sarcomas after undergoing primary tumor resection. J Pediatr Orthop2022;42(5):e501–e506; doi: 10.1097/bpo.0000000000002109
34.
CoreyAL, HaaseJE, AzzouzF, et al.Social support and symptom distress in adolescents/young adults with cancer. J Pediatr Oncol Nurs2008;25(5):275–284; doi: 10.1177/1043454208321117
35.
PennantS, LeeSC, HolmS, et al.The role of social support in adolescent/young adults coping with cancer treatment. Children (Basel)2019;7(1):2; doi: 10.3390/children7010002
36.
DonovanE, MartinSR, SeidmanLC, et al.A Mobile-based mindfulness and social support program for adolescents and young adults with sarcoma: Development and pilot testing. JMIR Mhealth Uhealth2019;7(3):e10921; doi: 10.2196/10921
37.
MorrisBA, LeporeSJ, WilsonB, et al.Adopting a survivor identity after cancer in a peer support context. J Cancer Surviv2014;8(3):427–436; doi: 10.1007/s11764-014-0355-5
38.
MorriM, BekkeringPW, CottiM, et al.Cross-cultural validation of the Italian version of the BT-DUX: A subjective measure of health-related quality of life in patients who underwent surgery for lower extremity malignant bone tumour. Cancers (Basel)2020;12(8):2015; doi: 10.3390/cancers12082015
39.
MorganSJ, FriedlyJL, AmtmannD, et al.Cross-sectional assessment of factors related to pain intensity and pain interference in lower limb prosthesis users. Arch Phys Med Rehabil2017;98(1):105–113; doi: 10.1016/j.apmr.2016.09.118
40.
JinJ, HongZ, RheaL, et al.PROMIS Physical Function and Pain Interference scores correlate with the lower extremity Toronto Extremity Salvage Score. JB JS Open Access2023;8(3):e23; doi: 10.2106/JBJS.OA.23.00011
41.
BasteckS, GuderWK, DirksenU, et al.Effects of an exercise intervention on gait function in young survivors of osteosarcoma with megaendoprosthesis of the lower extremity—Results from the pilot randomized controlled trial proGAIT. Curr Oncol2022;29(10):7754–7767; doi: 10.3390/curroncol29100613
42.
SaravananA, BajajP, MatthewsHL, et al.Fatigue and depressive mood in chronic low back pain. Pain Manag Nurs2024;25(3):225–230; doi: 10.1016/j.pmn.2024.02.003
43.
AlmeidaAM, LimaL, MartinsT. Understanding quality of life’s challenges in sarcoma patients: A mixed-methods study. Eur J Oncol Nurs2024;70:102585; doi: 10.1016/j.ejon.2024.102585
44.
KatsumotoS, OkamitsuM, YagoS. A qualitative literature review: Impact on life resulting from changes in physical function as perceived by adolescent young adult survivors of malignant bone tumors [in Japanese]. Bull Tokyo Healthc Univ2024;19:119–125.
