{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,5]],"date-time":"2026-06-05T04:05:42Z","timestamp":1780632342471,"version":"3.54.1"},"reference-count":17,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2026,3,11]],"date-time":"2026-03-11T00:00:00Z","timestamp":1773187200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2026,3,11]],"date-time":"2026-03-11T00:00:00Z","timestamp":1773187200000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["J. pediatr. gastroenterol. nutr."],"published-print":{"date-parts":[[2026,6]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Objectives<\/jats:title>\n                    <jats:p>Clinically, recurrent polyp growth is a characteristic feature of pediatric Peutz\u2013Jeghers syndrome (PJS) patients. However, the clinical characteristics of pediatric PJS patients grouped by postoperative recurrence time remain undefined. Furthermore, differences in clinical features between serine\/threonine kinase 11 (STK11)\u2010positive and \u2010negative patients, and the influence of STK11 mutation types on polyp recurrence time need to be elucidated. Our study aimed to characterize pediatric PJS based on postoperative polyp recurrence time and STK11 mutation status.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>We collected clinical data from 74 pediatric PJS patients diagnosed at Hunan Children's Hospital over the past decade. STK11 genomic profiling was performed using Sanger sequencing combined with multiplex ligation\u2010dependent probe amplification (MLPA) or whole exome sequencing (WES). Variables associated with gastrointestinal polyp recurrence were identified through least absolute shrinkage and selection operator (LASSO) regression, followed by multivariate logistic regression analysis of significant variables.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      All 74 pediatric PJS patients who experienced polyp recurrence post\u2010polypectomy were stratified by recurrence time (&gt;3, 1\u20133, and \u22641 year). Notably, 49.2% (31\/63) of STK11\u2010positive (STK11\n                      <jats:sup>pos<\/jats:sup>\n                      ) patients recurred within \u22641 year after polypectomy, while 81.8% (9\/11) of STK11\u2010negative (STK11\n                      <jats:sup>neg<\/jats:sup>\n                      ) patients with recurrence &gt;3 years after polypectomy. LASSO and multivariate logistic regression identified multiple jejunal polyps (odds ratio [OR]: 4.18, 95% confidence interval [CI]: 1.09\u201315.98) and giant small bowel polyps (OR: 4.06, 95% CI: 1.15\u201314.34) as independent risk factors for recurrence \u22641 year after polypectomy. Compared to STK11\n                      <jats:sup>neg<\/jats:sup>\n                      patients, STK11\n                      <jats:sup>pos<\/jats:sup>\n                      patients, especially when combined with a positive PJS family history exhibited significantly earlier symptom onset, higher gastrointestinal giant polyp burden, and higher polyp burden in the jejunum\/colon versus ileum. Analysis of 63 STK11\n                      <jats:sup>pos<\/jats:sup>\n                      patients revealed diverse mutation types\/sites and identified 15 novel pathogenic variants. STK11\n                      <jats:sup>pos<\/jats:sup>\n                      patients with de novo mutations exhibited a significantly higher incidence of hematochezia, along with a greater overall burden of giant polyps in the colon. No significant association was found between major mutation subtypes (frameshift, missense, deletion, and nonsense) and recurrence time, though missense mutations showed a trend toward earlier recurrence.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>This study reveals distinct clinical profiles across polyp recurrence intervals and between STK11\u2010positive and \u2010negative patients, while delineating the STK11 mutation landscape in pediatric PJS. These findings provide the genomic resource for pediatric PJS, offering critical insights into disease mechanisms and clinical management.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/jpn3.70376","type":"journal-article","created":{"date-parts":[[2026,3,11]],"date-time":"2026-03-11T10:01:56Z","timestamp":1773223316000},"page":"1377-1385","source":"Crossref","is-referenced-by-count":0,"title":["Distinct polyp recurrence timing and STK11 mutation status underlie clinical heterogeneity in pediatric Peutz\u2013Jeghers syndrome"],"prefix":"10.1002","volume":"82","author":[{"given":"Lingzhi","family":"Yuan","sequence":"first","affiliation":[{"name":"Department of Gastroenterology and Nutrition, the Affiliated Children's Hospital of Xiangya School of Medicine Central South University (Hunan Children's Hospital) Changsha China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Qin","family":"Tong","sequence":"additional","affiliation":[{"name":"Department of Gastroenterology and Nutrition, the Affiliated Children's Hospital of Xiangya School of Medicine Central South University (Hunan Children's Hospital) Changsha China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kang","family":"Xie","sequence":"additional","affiliation":[{"name":"Department of Gastroenterology and Nutrition, the Affiliated Children's Hospital of Xiangya School of Medicine Central South University (Hunan Children's Hospital) Changsha China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chengzhe","family":"Li","sequence":"additional","affiliation":[{"name":"The School of Pediatrics, Hengyang Medical School University of South China (Hunan Children's Hospital) Changsha China"},{"name":"Pediatrics Research Institute of Hunan Province the Affiliated Children's Hospital of Xiangya School of Medicine, Central South University (Hunan Children's Hospital) Changsha China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zhicheng","family":"Wang","sequence":"additional","affiliation":[{"name":"Department of Gastroenterology and Nutrition, the Affiliated Children's Hospital of Xiangya School of Medicine Central South University (Hunan Children's Hospital) Changsha China"},{"name":"Pediatrics Research Institute of Hunan Province the Affiliated Children's Hospital of Xiangya School of Medicine, Central South University (Hunan Children's Hospital) Changsha China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aomin","family":"Hong","sequence":"additional","affiliation":[{"name":"Department of Gastroenterology and Nutrition, the Affiliated Children's Hospital of Xiangya School of Medicine Central South University (Hunan Children's Hospital) Changsha China"},{"name":"Pediatrics Research Institute of Hunan Province the Affiliated Children's Hospital of Xiangya School of Medicine, Central South University (Hunan Children's Hospital) Changsha China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Liyuan","family":"Tang","sequence":"additional","affiliation":[{"name":"Department of Gastroenterology and Nutrition, the Affiliated Children's Hospital of Xiangya School of Medicine Central South University (Hunan Children's Hospital) Changsha China"},{"name":"Pediatrics Research Institute of Hunan Province the Affiliated Children's Hospital of Xiangya School of Medicine, Central South University (Hunan Children's Hospital) Changsha China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jieyu","family":"You","sequence":"additional","affiliation":[{"name":"Department of Gastroenterology and Nutrition, the Affiliated Children's Hospital of Xiangya School of Medicine Central South University (Hunan Children's Hospital) Changsha China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hongjuan","family":"Ouyang","sequence":"additional","affiliation":[{"name":"Department of Gastroenterology and Nutrition, the Affiliated Children's Hospital of Xiangya School of Medicine Central South University (Hunan Children's Hospital) Changsha China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Li","family":"Liu","sequence":"additional","affiliation":[{"name":"Department of Gastroenterology and Nutrition, the Affiliated Children's Hospital of Xiangya School of Medicine Central South University (Hunan Children's Hospital) Changsha China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wenting","family":"Zhang","sequence":"additional","affiliation":[{"name":"Department of Gastroenterology and Nutrition, the Affiliated Children's Hospital of Xiangya School of Medicine Central South University (Hunan Children's Hospital) Changsha China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Canlin","family":"Li","sequence":"additional","affiliation":[{"name":"Department of Gastroenterology and Nutrition, the Affiliated Children's Hospital of Xiangya School of Medicine Central South University (Hunan Children's Hospital) Changsha China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Meizheng","family":"Zhan","sequence":"additional","affiliation":[{"name":"Department of Gastroenterology and Nutrition, the Affiliated Children's Hospital of Xiangya School of Medicine Central South University (Hunan Children's Hospital) Changsha China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Na","family":"Jiang","sequence":"additional","affiliation":[{"name":"Department of Gastroenterology and Nutrition, the Affiliated Children's Hospital of Xiangya School of Medicine Central South University (Hunan Children's Hospital) Changsha China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jiaqi","family":"Duan","sequence":"additional","affiliation":[{"name":"Department of Gastroenterology and Nutrition, the Affiliated Children's Hospital of Xiangya School of Medicine Central South University (Hunan Children's Hospital) Changsha China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chenxi","family":"Liu","sequence":"additional","affiliation":[{"name":"Department of Gastroenterology and Nutrition, the Affiliated Children's Hospital of Xiangya School of Medicine Central South University (Hunan Children's Hospital) Changsha China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Juan","family":"Zhou","sequence":"additional","affiliation":[{"name":"Department of Gastroenterology and Nutrition, the Affiliated Children's Hospital of Xiangya School of Medicine Central South University (Hunan Children's Hospital) Changsha China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hua","family":"Wang","sequence":"additional","affiliation":[{"name":"Department of Medical Genetics the Affiliated Children's Hospital of Xiangya School of Medicine, Central South University (Hunan Children's Hospital) Changsha China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jinqiao","family":"Liu","sequence":"additional","affiliation":[{"name":"Department of Ultrasound the Affiliated Children's Hospital of Xiangya School of Medicine, Central South University (Hunan Children's Hospital) Changsha China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Guanghui","family":"Zhu","sequence":"additional","affiliation":[{"name":"The Affiliated Children's Hospital Of Xiangya School of Medicine, Central South University (Hunan Children's Hospital), Hunan Provincial Key Laboratory of Pediatric Orthopedics Changsha China"},{"name":"Furong Laboratory Changsha China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yafei","family":"Deng","sequence":"additional","affiliation":[{"name":"The School of Pediatrics, Hengyang Medical School University of South China (Hunan Children's Hospital) Changsha China"},{"name":"Pediatrics Research Institute of Hunan Province the Affiliated Children's Hospital of Xiangya School of Medicine, Central South University (Hunan Children's Hospital) Changsha China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3891-9851","authenticated-orcid":false,"given":"Hongmei","family":"Zhao","sequence":"additional","affiliation":[{"name":"Department of Gastroenterology and Nutrition, the Affiliated Children's Hospital of Xiangya School of Medicine Central South University (Hunan Children's Hospital) Changsha China"},{"name":"The School of Pediatrics, Hengyang Medical School University of South China (Hunan Children's Hospital) Changsha China"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,3,11]]},"reference":[{"issue":"4","key":"e_1_2_9_2_1","doi-asserted-by":"crossref","first-page":"447","DOI":"10.1007\/s00383-020-04621-3","article-title":"Polypoid disease of colon in children","volume":"36","author":"Soyer 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J","year":"2018","journal-title":"Fam Cancer"},{"issue":"10","key":"e_1_2_9_13_1","doi-asserted-by":"crossref","first-page":"1627","DOI":"10.3748\/wjg.v29.i10.1627","article-title":"Clinical features, diagnosis, and treatment of Peutz\u2010Jeghers syndrome: experience with 566 Chinese cases","volume":"29","author":"Xu ZX","year":"2023","journal-title":"World J Gastroenterol"},{"issue":"1","key":"e_1_2_9_14_1","doi-asserted-by":"crossref","first-page":"97","DOI":"10.1007\/s00384-009-0793-0","article-title":"Truncating mutations in Peutz\u2010Jeghers syndrome are associated with more polyps, surgical interventions and cancers","volume":"25","author":"Salloch H","year":"2010","journal-title":"Int J Colorectal Dis"},{"issue":"3","key":"e_1_2_9_15_1","doi-asserted-by":"crossref","first-page":"442","DOI":"10.1097\/MPG.0000000000002248","article-title":"Management of Peutz\u2010Jeghers syndrome in children and adolescents: a position paper from the ESPGHAN polyposis working group","volume":"68","author":"Latchford A","year":"2019","journal-title":"J Pediatr Gastroenterol Nutr"},{"issue":"7","key":"e_1_2_9_16_1","doi-asserted-by":"crossref","first-page":"606","DOI":"10.1016\/j.dld.2012.12.018","article-title":"Cancer risk associated with STK11\/LKB1 germline mutations in Peutz\u2010Jeghers syndrome patients: results of an Italian multicenter study","volume":"45","author":"Resta N","year":"2013","journal-title":"Dig Liver Dis"},{"key":"e_1_2_9_17_1","doi-asserted-by":"crossref","DOI":"10.1155\/2020\/9159315","article-title":"Clinical and genetic analyses of 38 chinese patients with Peutz\u2010Jeghers syndrome","volume":"2020","author":"Wu BD","year":"2020","journal-title":"BioMed Res Int"},{"issue":"2","key":"e_1_2_9_18_1","doi-asserted-by":"crossref","first-page":"199","DOI":"10.1097\/MPG.0000000000002166","article-title":"Clinical and genetic study of children with Peutz\u2010Jeghers syndrome identifies a high frequency of STK11 de novo mutation","volume":"68","author":"Zhao H","year":"2019","journal-title":"J Pediatr Gastroenterol Nutr"}],"container-title":["Journal of Pediatric Gastroenterology and 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