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When an event occurs in some patients prior to QOL score assessment, investigators may compare QOL scores between patient subgroups defined by the event after randomization. However, owing to postrandomization selection bias, this analysis can mislead investigators about treatment efficacy and result in paradoxical findings. The recent Japanese Osteoporosis Intervention Trial (JOINT\u201002), which compared the benefits of a combination therapy for fracture prevention with those of a monotherapy, exemplifies the case in point; the average QOL score was higher in the combination therapy arm for the unfractured subgroup but was lower for the fractured subgroup. To address this issue, principal strata effects (PSEs), which are treatment effects estimated within subgroups of individuals stratified by potential intermediate variable, have been discussed in the literature. In this paper, we describe a simple procedure for estimating the PSEs using marginal structural models. This procedure utilizes SAS code for the estimation. In addition, we present a simple sensitivity analysis method for examining the resulting estimates. The analyses of JOINT\u201002 data using these methods revealed that QOL scores were higher in the combination therapy arm than in the monotherapy arm for both subgroups.<\/jats:p>","DOI":"10.1002\/bimj.201800038","type":"journal-article","created":{"date-parts":[[2019,7,17]],"date-time":"2019-07-17T14:14:48Z","timestamp":1563372888000},"page":"1448-1461","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":4,"title":["Simple methods for the estimation and sensitivity analysis of principal strata effects using marginal structural models: Application to a bone fracture prevention trial"],"prefix":"10.1002","volume":"61","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-3013-7775","authenticated-orcid":false,"given":"Yukari","family":"Uemura","sequence":"first","affiliation":[{"name":"Biostatistics Section Department of Data Science Center for Clinical Sciences National Center for Global Health and Medicine  Shinjyuku\u2010ku Tokyo Japan"},{"name":"Biostatistics Division Clinical Research Support Center The University of Tokyo Hospital  Bunkyo\u2010ku Tokyo Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Masataka","family":"Taguri","sequence":"additional","affiliation":[{"name":"Department of Science Yokohama City University School of Data Science  Kanazawa\u2010ku Yokohama Japan"},{"name":"Research Center for Medical and Health Data Science The Institute of Statistical Mathematics  Tachikawa Tokyo Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Takuya","family":"Kawahara","sequence":"additional","affiliation":[{"name":"Biostatistics Division Clinical Research Support Center The University of Tokyo Hospital  Bunkyo\u2010ku Tokyo Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7261-2351","authenticated-orcid":false,"given":"Yasutaka","family":"Chiba","sequence":"additional","affiliation":[{"name":"Clinical Research Center Kindai University Hospital  Osakasayama Osaka Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2019,7,17]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1080\/01621459.1996.10476902"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.1002\/sim.4780132108"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.1002\/sim.1657"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cct.2009.10.003"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.1002\/bimj.201100085"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.4172\/2155-6180.1000e104"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1093\/aje\/kwq418"},{"key":"e_1_2_9_9_1","doi-asserted-by":"publisher","DOI":"10.1080\/01621459.2000.10474210"},{"key":"e_1_2_9_10_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00198-004-1705-3"},{"key":"e_1_2_9_11_1","doi-asserted-by":"publisher","DOI":"10.1093\/biostatistics\/kxl027"},{"key":"e_1_2_9_12_1","doi-asserted-by":"publisher","DOI":"10.1093\/biostatistics\/kxp060"},{"key":"e_1_2_9_13_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.0006-341X.2002.00021.x"},{"key":"e_1_2_9_14_1","doi-asserted-by":"publisher","DOI":"10.1093\/aje\/kwq439"},{"key":"e_1_2_9_15_1","doi-asserted-by":"publisher","DOI":"10.1002\/sim.3533"},{"key":"e_1_2_9_16_1","doi-asserted-by":"publisher","DOI":"10.1111\/1541-0420.00063"},{"key":"e_1_2_9_17_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1541-0420.2008.01014.x"},{"key":"e_1_2_9_18_1","doi-asserted-by":"publisher","DOI":"10.1097\/01.EDE.0000042804.12056.6C"},{"key":"e_1_2_9_19_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00198-008-0761-5"},{"key":"e_1_2_9_20_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.0006-341X.2005.030227.x"},{"key":"e_1_2_9_21_1","doi-asserted-by":"publisher","DOI":"10.1097\/00001648-200009000-00012"},{"key":"e_1_2_9_22_1","doi-asserted-by":"publisher","DOI":"10.1136\/jech.2002.006361"},{"key":"e_1_2_9_23_1","unstructured":"ICH E9(R1). 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