{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,4]],"date-time":"2026-05-04T06:52:29Z","timestamp":1777877549207,"version":"3.51.4"},"reference-count":28,"publisher":"SAGE Publications","license":[{"start":{"date-parts":[[2026,3,1]],"date-time":"2026-03-01T00:00:00Z","timestamp":1772323200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"},{"start":{"date-parts":[[2026,3,1]],"date-time":"2026-03-01T00:00:00Z","timestamp":1772323200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"funder":[{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"publisher","award":["81772086, 82072177"],"award-info":[{"award-number":["81772086, 82072177"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"publisher"}]},{"name":"Shanghai Municipal Key Clinical Specialty","award":["shslczdzk00901"],"award-info":[{"award-number":["shslczdzk00901"]}]}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["Journal of Otolaryngology - Head &amp; Neck Surgery"],"published-print":{"date-parts":[[2026,3]]},"abstract":"<jats:sec>\n                    <jats:title>Importance<\/jats:title>\n                    <jats:p>Idiopathic hypogonadotropic hypogonadism (IHH) associated with congenital hemi-arrhinia is rare but can substantially impact patients\u2019 quality of life. The underlying genetic etiology remains to be clarified, and surgical approaches for reconstructing congenital hemi-arrhinia are sparsely reported.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>To provide a reference for clinical diagnosis and management by reporting the patient\u2019s genetic findings and surgical outcomes.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Design<\/jats:title>\n                    <jats:p>Case report.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Setting<\/jats:title>\n                    <jats:p>Tertiary public referral hospital.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Participants<\/jats:title>\n                    <jats:p>A patient diagnosed with IHH and congenital hemi-arrhinia.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Intervention or Exposures<\/jats:title>\n                    <jats:p>Whole-exome sequencing was performed, and the patient\u2019s nose was reconstructed using the contralateral nasal dorsum flap as the inner lining, an L-strut of rib and costal cartilage graft as the structural support, and a pre-expanded forehead flap as the outer skin envelope.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Main Outcome Measures<\/jats:title>\n                    <jats:p>One-year postoperative outcomes were evaluated using the Rhinoplasty Outcome Evaluation (ROE) and Nasal Obstruction Symptom Evaluation (NOSE) scales.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Whole-exome sequencing revealed maternally and paternally inherited missense mutations in Gonadotropin-Releasing Hormone Receptor (GNRHR) (c.719G\u2009&gt;\u2009A) and KISS1 (c.58G\u2009&gt;\u2009A), respectively. One-year follow-up demonstrated stable nasal reconstruction, with the ROE score improving from 4 to 17 out of 24. The NOSE score remained 1 out of 20 pre- and postoperatively, indicating no significant change in nasal airflow perception.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>The 4-stage surgical procedure is a viable and effective reconstructive option for hemi-arrhinia.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Relevance<\/jats:title>\n                    <jats:p>Our genetic findings may serve as a reference for investigating the underlying causes of IHH. The described surgical approach provides a practical reference for surgeons performing hemi-nose reconstruction.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1177\/19160216261425177","type":"journal-article","created":{"date-parts":[[2026,3,28]],"date-time":"2026-03-28T09:40:03Z","timestamp":1774690803000},"update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":0,"title":["Idiopathic Hypogonadotropic Hypogonadism with a Rare Hemi-Arrhinia: A Case Report with Nasal Reconstruction"],"prefix":"10.1177","volume":"55","author":[{"given":"Yunhan","family":"Liu","sequence":"first","affiliation":[{"name":"Department of Plastic and Reconstructive Surgery, Shanghai Ninth People\u2019s Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yimin","family":"Khoong","sequence":"additional","affiliation":[{"name":"Department of Plastic and Reconstructive Surgery, Shanghai Ninth People\u2019s Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Xin","family":"Huang","sequence":"additional","affiliation":[{"name":"Department of Plastic and Reconstructive Surgery, Shanghai Ninth People\u2019s Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shuchen","family":"Gu","sequence":"additional","affiliation":[{"name":"Department of Plastic and Reconstructive Surgery, Shanghai Ninth People\u2019s Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tao","family":"Zan","sequence":"additional","affiliation":[{"name":"Department of Plastic and Reconstructive Surgery, Shanghai Ninth People\u2019s Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"179","published-online":{"date-parts":[[2026,3,28]]},"reference":[{"key":"e_1_3_3_2_2","doi-asserted-by":"publisher","DOI":"10.1038\/nrendo.2015.112"},{"key":"e_1_3_3_3_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.metabol.2017.10.012"},{"key":"e_1_3_3_4_2","first-page":"113","article-title":"Update on the genetics of idiopathic hypogonadotropic hypogonadism","volume":"9","author":"Topalo\u011flu AK.","year":"2017","unstructured":"Topalo\u011flu AK. Update on the genetics of idiopathic hypogonadotropic hypogonadism. J Clin Res Pediatr Endocrinol. 2017;9(Suppl. 2):113-122.","journal-title":"J Clin Res Pediatr Endocrinol"},{"key":"e_1_3_3_5_2","doi-asserted-by":"publisher","DOI":"10.1210\/clinem\/dgaa065"},{"key":"e_1_3_3_6_2","doi-asserted-by":"publisher","DOI":"10.1080\/02844310802517259"},{"key":"e_1_3_3_7_2","doi-asserted-by":"publisher","DOI":"10.1097\/SCS.0000000000005091"},{"key":"e_1_3_3_8_2","doi-asserted-by":"publisher","DOI":"10.1136\/bcr-2021-245424"},{"key":"e_1_3_3_9_2","doi-asserted-by":"publisher","DOI":"10.1097\/SCS.0000000000006522"},{"key":"e_1_3_3_10_2","doi-asserted-by":"publisher","DOI":"10.1097\/SCS.0000000000004803"},{"key":"e_1_3_3_11_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.bjps.2008.08.040"},{"key":"e_1_3_3_12_2","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM199711273372205"},{"key":"e_1_3_3_13_2","doi-asserted-by":"publisher","DOI":"10.21037\/tau-20-1390"},{"key":"e_1_3_3_14_2","doi-asserted-by":"publisher","DOI":"10.21037\/tau.2020.03.33"},{"key":"e_1_3_3_15_2","doi-asserted-by":"publisher","DOI":"10.1111\/andr.12563"},{"key":"e_1_3_3_16_2","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2012-1264"},{"key":"e_1_3_3_17_2","doi-asserted-by":"publisher","DOI":"10.3389\/fendo.2022.925206"},{"key":"e_1_3_3_18_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.bjps.2004.01.007"},{"key":"e_1_3_3_19_2","doi-asserted-by":"publisher","DOI":"10.1007\/s12070-007-0017-2"},{"key":"e_1_3_3_20_2","doi-asserted-by":"publisher","DOI":"10.1097\/SCS.0000000000000542"},{"key":"e_1_3_3_21_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.bjps.2008.02.039"},{"key":"e_1_3_3_22_2","doi-asserted-by":"publisher","DOI":"10.1097\/SCS.0000000000000518"},{"key":"e_1_3_3_23_2","doi-asserted-by":"publisher","DOI":"10.1097\/MOO.0000000000000573"},{"key":"e_1_3_3_24_2","doi-asserted-by":"publisher","DOI":"10.1097\/01.PRS.0000131870.52649.98"},{"key":"e_1_3_3_25_2","doi-asserted-by":"publisher","DOI":"10.1097\/PRS.0b013e3181d0acb1"},{"key":"e_1_3_3_26_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.bjps.2009.05.005"},{"key":"e_1_3_3_27_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.bjorl.2020.07.001"},{"key":"e_1_3_3_28_2","doi-asserted-by":"publisher","DOI":"10.1097\/PRS.0b013e3181c82f12"},{"key":"e_1_3_3_29_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.bjps.2018.03.017"}],"container-title":["Journal of Otolaryngology - Head &amp; Neck Surgery"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/19160216261425177","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/full-xml\/10.1177\/19160216261425177","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/19160216261425177","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,4,30]],"date-time":"2026-04-30T22:54:37Z","timestamp":1777589677000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.sagepub.com\/doi\/10.1177\/19160216261425177"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,3]]},"references-count":28,"alternative-id":["10.1177\/19160216261425177"],"URL":"https:\/\/doi.org\/10.1177\/19160216261425177","relation":{},"ISSN":["1916-0216","1916-0216"],"issn-type":[{"value":"1916-0216","type":"print"},{"value":"1916-0216","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,3]]},"article-number":"19160216261425177"}}