{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,1]],"date-time":"2026-09-01T23:50:38Z","timestamp":1788306638777,"version":"build-2803163510"},"reference-count":40,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2015,12,1]],"date-time":"2015-12-01T00:00:00Z","timestamp":1448928000000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/www.springer.com\/tdm"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Head Face Med"],"published-print":{"date-parts":[[2015,12]]},"DOI":"10.1186\/s13005-015-0095-4","type":"journal-article","created":{"date-parts":[[2015,12,28]],"date-time":"2015-12-28T11:06:30Z","timestamp":1451300790000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":8,"title":["CANDLE SYNDROME: Orofacial manifestations and dental implications"],"prefix":"10.1186","volume":"11","author":[{"given":"T.","family":"Roberts","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"L.","family":"Stephen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"C.","family":"Scott","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"T.","family":"di Pasquale","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"A.","family":"Naser-eldin","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"M.","family":"Chetty","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"S.","family":"Shaik","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"L.","family":"Lewandowski","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"P.","family":"Beighton","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2015,12,28]]},"reference":[{"key":"95_CR1","first-page":"703","volume":"24","author":"S Yamada","year":"1984","unstructured":"Yamada S, Toyoshima I, Mori S, Tsubaki T. Sibling cases with lipodystrophic skin change, muscular atrophy, recurrent skin eruptions, and deformities and contractures of the joints: a possible new clinical entity. Rinsho Shinkeigaku. 1984;24:703\u201310.","journal-title":"Rinsho Shinkeigaku"},{"key":"95_CR2","first-page":"77","volume":"45","author":"A Nakajo","year":"1939","unstructured":"Nakajo A. Secondary hypertrophic osteoperiostosis with pernio. J Derm Urol. 1939;45:77\u201386.","journal-title":"J Derm Urol."},{"key":"95_CR3","first-page":"136","volume":"60","author":"N Nishimura","year":"1950","unstructured":"Nishimura N, Deki T, Kato S. Hypertrophic pulmonary osteo-arthropathy with pernio-like eruption in the two families: report of the three cases. Jpn J Derm Venereol. 1950;60:136\u201341.","journal-title":"Jpn J Derm Venereol"},{"key":"95_CR4","doi-asserted-by":"publisher","first-page":"1053","DOI":"10.1001\/archderm.1985.01660080107026","volume":"121","author":"Y Kitano","year":"1985","unstructured":"Kitano Y, Matsunaga E, Morimoto T, Okada N, Sano S. A syndrome with nodular erythema, elongated and thickened fingers, and emaciation. Arch Derm. 1985;121:1053\u20136.","journal-title":"Arch Derm"},{"key":"95_CR5","doi-asserted-by":"publisher","first-page":"42","DOI":"10.2169\/internalmedicine.32.42","volume":"32","author":"M Tanaka","year":"1993","unstructured":"Tanaka M, Miyatani N, Yamada S, Miyashita K, Toyoshima I, Sakuma K, et al. Hereditary lipo-muscular atrophy with joint contracture, skin eruptions and hyper-gamma-globulinemia: a new syndrome. Intern Med. 1993;32:42\u20135.","journal-title":"Intern Med"},{"key":"95_CR6","doi-asserted-by":"publisher","first-page":"14914","DOI":"10.1073\/pnas.1106015108","volume":"108","author":"K Arima","year":"2011","unstructured":"Arima K, Kinoshita A, Mishima H, Kanazawa N, Kaneko T, Mizushima T, et al. Proteasome assembly defect due to a proteasome subunit beta type 8 (PSMB8) mutation causes the autoinflammatory disorder, Nakajo-Nishimura syndrome. Proc Nat Acad Sci. 2011;108:14914\u20139. doi: 10.1073\/pnas.1106015108 .","journal-title":"Proc Nat Acad Sci"},{"key":"95_CR7","doi-asserted-by":"publisher","first-page":"E58","DOI":"10.1210\/jc.2010-0488","volume":"95","author":"A Garg","year":"2010","unstructured":"Garg A, Hernandez MD, Sousa AB, Subramanyam L, Martinez de Villarreal L, dos Santos HG, et al. An autosomal recessive syndrome of joint contractures, muscular atrophy, microcytic anemia, and panniculitis-associated lipodystrophy. J Clin Endocr Metab. 2010;95:E58\u201363. doi: 10.1210\/jc.2010-0488 .","journal-title":"J Clin Endocr Metab"},{"key":"95_CR8","doi-asserted-by":"publisher","first-page":"489","DOI":"10.1016\/j.jaad.2009.04.046","volume":"62","author":"A Torrelo","year":"2010","unstructured":"Torrelo A, Patel S, Colmenero I, Gurbindo D, Lendinez F, Hernandez A, et al. Chronic atypical neutrophilic dermatosis with lipodystrophy and elevated temperature (CANDLE) syndrome. J Am Acad Derm. 2010;62:489\u201395. doi: 10.1016\/j.jaad.2009.04.046 .","journal-title":"J Am Acad Derm"},{"key":"95_CR9","doi-asserted-by":"publisher","unstructured":"Wang H, Das L, Tan Hung Tiong J, Vasanwala RF, Arkachaisri T. CANDLE syndrome: an extended clinical spectrum. Rheumatology 2014;53:2120-2122 doi: 10.1093\/rheumatology\/keu297 .","DOI":"10.1093\/rheumatology\/keu297"},{"key":"95_CR10","volume-title":"WHO scientific group on the assessment of osteoporosis at primary health care level","author":"WHO","year":"2004","unstructured":"WHO. WHO scientific group on the assessment of osteoporosis at primary health care level. Geneva: WHO Press; 2004."},{"key":"95_CR11","doi-asserted-by":"publisher","first-page":"774","DOI":"10.1016\/0030-4220(93)90438-A","volume":"75","author":"E Klemetti","year":"1993","unstructured":"Klemetti E, Kolmakov S, Heiskanen P, Vainio P, Lassila V. Panoramic mandibular index and bone mineral densities in postmenopausal women. Oral Surg Oral Med Oral. 1993;75:774\u20139.","journal-title":"Oral Surg Oral Med Oral"},{"key":"95_CR12","doi-asserted-by":"publisher","first-page":"847","DOI":"10.1016\/j.cden.2012.07.010","volume":"56","author":"H Devlin","year":"2012","unstructured":"Devlin H. Identification of the risk for osteoporosis in dental patients. Dent Clin N Am. 2012;56:847\u201361. doi: 10.1016\/j.cden.2012.07.010 .","journal-title":"Dent Clin N Am"},{"issue":"6","key":"95_CR13","doi-asserted-by":"publisher","first-page":"473","DOI":"10.1097\/MAJ.0b013e3182972148","volume":"346","author":"S Mansour","year":"2013","unstructured":"Mansour S, AlGhamdi AS, Javed F, Marzouk H, Khan EA. Panoramic radiomorphometric indices as reliable parameters in predicting osteoporosis. Am J Med Sci. 2013;346(6):473\u20138. doi: 10.1097\/MAJ.0b013e3182972148 .","journal-title":"Am J Med Sci"},{"key":"95_CR14","doi-asserted-by":"publisher","first-page":"405","DOI":"10.1177\/00220345600390022301","volume":"39","author":"WS Hunter","year":"1960","unstructured":"Hunter WS, Priest WR. Errors and discrepancies in measurement of tooth size. J Dent Res. 1960;39:405\u201314.","journal-title":"J Dent Res"},{"key":"95_CR15","doi-asserted-by":"publisher","first-page":"866","DOI":"10.1016\/j.ajhg.2010.10.031","volume":"87","author":"AK Agarwal","year":"2010","unstructured":"Agarwal AK, Xing C, DeMartino GN, Mizrachi D, Hernandez MD, Sousa AB, et al. PSMB8 encoding the beta-5i proteasome subunit is mutated in joint contractures, muscle atrophy, microcytic anemia, and panniculitis-induced lipodystrophy syndrome. Am J Hum Genet. 2010;87:866\u201372. doi: 10.1016\/j.ajhg.2010.10.031 .","journal-title":"Am J Hum. Genet"},{"key":"95_CR16","doi-asserted-by":"publisher","first-page":"4150","DOI":"10.1172\/JCI58414","volume":"121","author":"A Kitamura","year":"2011","unstructured":"Kitamura A, Maekawa Y, Uehara H, Izumi K, Kawachi I, Nishizawa M, et al. A mutation in the immunoproteasome subunit PSMB8 causes autoinflammation and lipodystrophy in humans. J Clin Invest. 2011;121:4150\u201360. doi: 10.1172\/JCI58414 .","journal-title":"J Clin Invest"},{"key":"95_CR17","doi-asserted-by":"crossref","unstructured":"Cavalcante MP, Brunelli JB, Miranda CC, Novak GV, Malle L, Aikawa NE, et al. CANDLE syndrome: chronic atypical neutrophilic dermatosis with lipodystrophy and elevated temperature-a rare case with a novel mutation. Eur J Pediatr. 2015. [Epub ahead of print].","DOI":"10.1007\/s00431-015-2668-4"},{"issue":"3","key":"95_CR18","doi-asserted-by":"publisher","first-page":"895","DOI":"10.1002\/art.33368","volume":"64","author":"Y Liu","year":"2012","unstructured":"Liu Y, Ramot Y, Torrelo A, Paller AS, et al. Mutations in proteasome subunit \u03b2 type 8 cause chronic atypical neutrophilic dermatosis with lipodystrophy and elevated temperature with evidence of genetic and phenotypic heterogeneity. Arthritis Rheum. 2012;64(3):895\u2013907. doi: 10.1002\/art.33368 .","journal-title":"Arthritis Rheum"},{"issue":"11","key":"95_CR19","doi-asserted-by":"publisher","first-page":"4196","DOI":"10.1172\/JCI81260","volume":"125","author":"A Brehm","year":"2015","unstructured":"Brehm A, Liu Y, Sheikh A. et. Additive loss-of-function proteasome subunit mutations in CANDLE\/PRAAS patients promote type I IFN production. J Clin Invest. 2015;125(11):4196\u2013211. doi: 10.1172\/JCI81260 . Epub 2015 Oct 20.","journal-title":"J Clin Invest"},{"key":"95_CR20","first-page":"114","volume-title":"Developmental anomalies and eruption disturbances and some acquired disorders of the teeth. In: Oral pathology","author":"I Waal Van der","year":"1988","unstructured":"Van der Waal I, Van der Kwast WAM. Developmental anomalies and eruption disturbances and some acquired disorders of the teeth. In: Oral pathology. Chicago: Quintessence Publishing Co Inc; 1988. p. 114."},{"key":"95_CR21","series-title":"A Textbook of Oral Pathology","first-page":"37","volume-title":"Develpmental Disturbances of oral and paraoral structures","author":"WG Shafer","year":"1983","unstructured":"Shafer WG, Hine MK, Levy BM. Develpmental Disturbances of oral and paraoral structures, A Textbook of Oral Pathology. Philadelphia: W. B. Saunders Co; 1983. p. 37."},{"issue":"7","key":"95_CR22","doi-asserted-by":"publisher","first-page":"918","DOI":"10.1016\/j.archoralbio.2012.01.005","volume":"57","author":"G Galluccio","year":"2012","unstructured":"Galluccio G, Castellano M, La Monaca C. Genetic basis of non-syndromic anomalies of human tooth number. Arch Oral Biol. 2012;57(7):918\u201330. doi: 10.1016\/j.archoralbio.2012.01.005 .","journal-title":"Arch Oral Biol"},{"issue":"Suppl 1","key":"95_CR23","doi-asserted-by":"publisher","first-page":"3","DOI":"10.1016\/j.archoralbio.2009.09.005","volume":"54","author":"AH Brook","year":"2009","unstructured":"Brook AH. Multilevel complex interactions between genetic, epigenetic and environmental factors in the aetiology of anomalies of dental development. Arch Oral Biol. 2009;54 Suppl 1:3\u201317. doi: 10.1016\/j.archoralbio.2009.09.005 .","journal-title":"Arch Oral Biol"},{"key":"95_CR24","doi-asserted-by":"publisher","first-page":"125","DOI":"10.1038\/bdj.2008.47","volume":"204","author":"MJ Garnett","year":"2008","unstructured":"Garnett MJ, Nohl FS, Barclay SC. Management of patients with reduced oral aperture and mandibular hypomobility (trismus) and implications for operative dentistry. BMJ. 2008;204:125\u201331. doi: 10.1038\/bdj.2008.47 .","journal-title":"BMJ"},{"issue":"2","key":"95_CR25","first-page":"167","volume":"55","author":"S Steigman","year":"1985","unstructured":"Steigman S, Weissberg Y. Spaced dentition. An epidemiologic study. Angle Orthod. 1985;55(2):167\u201376.","journal-title":"Angle Orthod"},{"issue":"3","key":"95_CR26","doi-asserted-by":"publisher","first-page":"209","DOI":"10.1111\/j.1708-8240.2009.00261.x","volume":"21","author":"LW Boushell","year":"2009","unstructured":"Boushell LW. Diastema. J Esthet Restor Dent. 2009;21(3):209\u201310. doi: 10.1111\/j.1708-8240.2009.00261.x .","journal-title":"J Esthet Restor Dent"},{"issue":"6","key":"95_CR27","doi-asserted-by":"publisher","first-page":"970","DOI":"10.1016\/j.mehy.2011.08.022","volume":"77","author":"AC Gallup","year":"2011","unstructured":"Gallup AC, Hack GD. Human paranasal sinuses and selective brain cooling: a ventilation system activated by yawning? Med Hypotheses. 2011;77(6):970\u20131003. doi: 10.1016\/j.mehy.2011.08.022 .","journal-title":"Med Hypotheses"},{"issue":"4","key":"95_CR28","doi-asserted-by":"publisher","first-page":"518","DOI":"10.1002\/ajmg.1320440428","volume":"44","author":"PF Ippel","year":"1992","unstructured":"Ippel PF, Gorlin RJ, Lenz W, van Doorne JM, Bijlsma JB. Craniofacial dysostosis, hypertrichosis, genital hypoplasia, ocular, dental, and digital defects: confirmation of the Gorlin-Chaudhry-Moss syndrome. Am J Med Genet. 1992;44(4):518.","journal-title":"Am J Med Genet"},{"issue":"Suppl 1","key":"95_CR29","doi-asserted-by":"publisher","first-page":"41","DOI":"10.4103\/0976-237X.95103","volume":"3","author":"P Poornima","year":"2012","unstructured":"Poornima P, Patil P, Subbareddy V, Arora G. Dentofacial characteristics in William's syndrome. Contemp Clin Dent. 2012;3 Suppl 1:41\u2013S44. doi: 10.4103\/0976-237X.95103 .","journal-title":"Contemp Clin Dent"},{"key":"95_CR30","doi-asserted-by":"publisher","first-page":"58","DOI":"10.1016\/j.ics.2006.06.001","volume":"1298","author":"DF Gunther","year":"2006","unstructured":"Gunther DF, Sybert VP. Lymphatic, tooth and skin manifestations in Turners syndrome. Int Congr Series. 2006;1298:58\u201362.","journal-title":"Int Congr Series"},{"issue":"4","key":"95_CR31","doi-asserted-by":"publisher","first-page":"479","DOI":"10.1067\/moe.2002.127584","volume":"94","author":"MC Hayta\u00e7","year":"2002","unstructured":"Hayta\u00e7 MC, Oztun\u00e7 H, Mete UO, Kaya M. Rothmund-Thomson syndrome: a case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2002;94(4):479\u201384.","journal-title":"Oral Surg Oral Med Oral Pathol Oral Radiol Endod"},{"key":"95_CR32","doi-asserted-by":"publisher","unstructured":"Canger EM, Celenk P, Devrim I, Av\u015far A. Oral findings of Rothmund-Thomson syndrome. Case Rep Dent . 2013;2013:935716. doi: 10.1155\/2013\/935716 .","DOI":"10.1155\/2013\/935716"},{"issue":"3","key":"95_CR33","doi-asserted-by":"publisher","first-page":"305","DOI":"10.17796\/jcpd.26.3.l02834m2827m0132","volume":"26","author":"F Seymen","year":"2002","unstructured":"Seymen F, Tuna B, Kayserili H. Seckel syndrome: report of a case. J Clin Pediatr Dent. 2002;26(3):305\u20139.","journal-title":"J Clin Pediatr Dent"},{"issue":"10","key":"95_CR34","doi-asserted-by":"publisher","first-page":"639","DOI":"10.1111\/j.1600-0714.2006.00462.x","volume":"35","author":"PJ Coster De","year":"2006","unstructured":"De Coster PJ, Verbeeck RM, Holthaus V, Martens LC, Vral A. Seckel syndrome associated with oligodontia, microdontia, enamel hypoplasia, delayed eruption, and dentin demineralization: a new variant? J Oral Pathol Med. 2006;35(10):639\u201341.","journal-title":"J Oral Pathol Med"},{"issue":"4","key":"95_CR35","doi-asserted-by":"publisher","first-page":"413","DOI":"10.1002\/ajmg.a.20606","volume":"126A","author":"A Rajab","year":"2004","unstructured":"Rajab A, Kunze J, Mundlos S. Spondyloepiphyseal dysplasia Omani type: a new recessive type of SED with progressive spinal involvement. Am J Med Genet A. 2004;126A(4):413\u20139.","journal-title":"Am J Med Genet A"},{"key":"95_CR36","doi-asserted-by":"publisher","first-page":"685781","DOI":"10.1155\/2013\/685781","volume":"2013","author":"A Singhal","year":"2013","unstructured":"Singhal A, Singhal P, Gupta R, Jarial KD. True generalized microdontia and hypodontia with spondyloepiphyseal dysplasia. Case Rep Dent. 2013;2013:685781. doi: 10.1155\/2013\/685781 .","journal-title":"Case Rep Dent"},{"issue":"8\u20139","key":"95_CR37","doi-asserted-by":"publisher","first-page":"441","DOI":"10.1016\/j.ejmg.2012.03.005","volume":"55","author":"Y Moussaid","year":"2012","unstructured":"Moussaid Y, Griffiths D, Richard B, Dieux A, Lemerrer M, L\u00e9ger J, et al. Oral manifestations of patients with Kenny-Caffey Syndrome. Eur J Med Genet. 2012;55(8\u20139):441\u20135. doi: 10.1016\/j.ejmg.2012.03.005 .","journal-title":"Eur J Med Genet"},{"issue":"2","key":"95_CR38","doi-asserted-by":"publisher","first-page":"154","DOI":"10.1111\/j.1365-263X.2011.01160.x","volume":"22","author":"J Norderyd","year":"2012","unstructured":"Norderyd J, Aronsson J. Hypoplastic root cementum and premature loss of primary teeth in Coffin-Lowry syndrome: a case report. Int J Paediatr Dent. 2012;22(2):154\u20136. doi: 10.1111\/j.1365-263X.2011.01160.x .","journal-title":"Int J Paediatr Dent"},{"issue":"2","key":"95_CR39","doi-asserted-by":"publisher","first-page":"136","DOI":"10.1002\/ajmg.1320580209","volume":"58","author":"HJ Lin","year":"1995","unstructured":"Lin HJ, Sue GY, Berkowitz CD, Brasel JA, Lachman RS. Microdontia with severe microcephaly and short stature in two brothers: osteodysplastic primordial dwarfism with dental findings. Am J Med Genet. 1995;58(2):136\u201342.","journal-title":"Am J Med Genet"},{"issue":"2","key":"95_CR40","doi-asserted-by":"publisher","first-page":"181","DOI":"10.1002\/ajmg.a.30079","volume":"130A","author":"PN Kantaputra","year":"2004","unstructured":"Kantaputra PN, Tanpaiboon P, Unachak K, Praphanphoj V. Microcephalic osteodysplastic primordial dwarfism with severe microdontia and skin anomalies: confirmation of a new syndrome. Am J Med Genet A. 2004;130A(2):181\u201390.","journal-title":"Am J Med Genet A"}],"container-title":["Head &amp; Face Medicine"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/link.springer.com\/content\/pdf\/10.1186\/s13005-015-0095-4.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/link.springer.com\/article\/10.1186\/s13005-015-0095-4\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/link.springer.com\/content\/pdf\/10.1186\/s13005-015-0095-4.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2019,9,3]],"date-time":"2019-09-03T00:12:12Z","timestamp":1567469532000},"score":1,"resource":{"primary":{"URL":"http:\/\/head-face-med.biomedcentral.com\/articles\/10.1186\/s13005-015-0095-4"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2015,12]]},"references-count":40,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2015,12]]}},"alternative-id":["95"],"URL":"https:\/\/doi.org\/10.1186\/s13005-015-0095-4","relation":{},"ISSN":["1746-160X"],"issn-type":[{"value":"1746-160X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2015,12]]},"article-number":"38"}}