{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,17]],"date-time":"2026-08-17T20:44:51Z","timestamp":1786999491433,"version":"build-2736575974"},"reference-count":24,"publisher":"Cambridge University Press (CUP)","issue":"10","license":[{"start":{"date-parts":[[2018,8,13]],"date-time":"2018-08-13T00:00:00Z","timestamp":1534118400000},"content-version":"unspecified","delay-in-days":0,"URL":"https:\/\/www.cambridge.org\/core\/terms"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["J. Laryngol. Otol."],"published-print":{"date-parts":[[2018,10]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec id=\"S0022215118001329_sec_a1\">\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Lipoid proteinosis is a rare autosomal recessive disorder caused by mutations in the extracellular matrix protein 1 gene. It is characterised by deposition of hyaline material in the skin and mucous membranes. This paper describes the management of two cases with laryngopharyngeal disease.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec id=\"S0022215118001329_sec_a2\" sec-type=\"methods\">\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>Two patients with a biopsy diagnosis of lipoid proteinosis were identified from the surgical pathology archive covering the period 2004\u20132016. Their notes were reviewed.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec id=\"S0022215118001329_sec_a3\" sec-type=\"results\">\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>An adult male and an adult female were identified. Both had dysphonia and laryngopharyngeal lesions. The patients underwent interval laser microlaryngoscopy to debulk disease but minimise mucosal injury and scarring, using a \u2018pepper pot\u2019 technique. Both had adequate symptom control.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec id=\"S0022215118001329_sec_a4\" sec-type=\"conclusion\">\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>Lipoid proteinosis is a rare genetic condition, which typically presents in infancy with dysphonia and subsequent skin involvement. Two cases are presented to demonstrate that laryngotracheal symptoms can be controlled with interval laser debulking and the \u2018pepper pot\u2019 technique without causing stenosis.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1017\/s0022215118001329","type":"journal-article","created":{"date-parts":[[2018,8,13]],"date-time":"2018-08-13T01:50:31Z","timestamp":1534125031000},"page":"936-939","source":"Crossref","is-referenced-by-count":7,"title":["The management of laryngeal lipoid proteinosis"],"prefix":"10.1017","volume":"132","author":[{"given":"M","family":"Ally","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"A J","family":"Kinshuck","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"A","family":"Sandison","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"G S","family":"Sandhu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"56","published-online":{"date-parts":[[2018,8,13]]},"reference":[{"key":"S0022215118001329_ref22","first-page":"245","article-title":"Sarcoidosis of the larynx treated with CO2 laser","volume":"14","author":"Ruff","year":"1985","journal-title":"J Otolaryngol"},{"key":"S0022215118001329_ref14","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2230.2002.01143.x"},{"key":"S0022215118001329_ref24","doi-asserted-by":"publisher","DOI":"10.1288\/00005537-198908001-00001"},{"key":"S0022215118001329_ref17","doi-asserted-by":"publisher","DOI":"10.1017\/S0022215100095141"},{"key":"S0022215118001329_ref13","doi-asserted-by":"crossref","first-page":"E30","DOI":"10.1177\/014556131509400304","article-title":"Lipoid proteinosis of the larynx","volume":"94","author":"Virk","year":"2015","journal-title":"Ear Nose Throat J"},{"key":"S0022215118001329_ref8","doi-asserted-by":"crossref","first-page":"148","DOI":"10.4103\/2229-5178.96723","article-title":"Lipoid proteinosis: histopathological characterization of early papulovesicular lesions","volume":"3","author":"Gutte","year":"2012","journal-title":"Indian Dermatol Online J"},{"key":"S0022215118001329_ref12","doi-asserted-by":"publisher","DOI":"10.1001\/archoto.2010.16"},{"key":"S0022215118001329_ref7","doi-asserted-by":"publisher","DOI":"10.1016\/0165-5876(88)90048-1"},{"key":"S0022215118001329_ref1","first-page":"162","article-title":"Lipoid proteinosis: case report and review of the literature","volume":"26","author":"Di Giandomenico","year":"2006","journal-title":"Acta Otorhinolaryngol Ital"},{"key":"S0022215118001329_ref10","first-page":"734","article-title":"Radiographic and computed tomographic findings in lipid proteinosis. A case report","volume":"65","author":"Friedman","year":"1984","journal-title":"S Afr Med J"},{"key":"S0022215118001329_ref5","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijporl.2004.07.012"},{"key":"S0022215118001329_ref3","first-page":"6","article-title":"Lipoid proteinosis in a child with recurrent respiratory infection","volume":"3","author":"Kumar","year":"2009","journal-title":"EJ Indian Soc Teledermatol"},{"key":"S0022215118001329_ref9","first-page":"97","article-title":"Eyelid beading - a useful diagnostic clue for lipoid proteinosis","volume":"38","author":"Thappa","year":"2001","journal-title":"Indian Pediatr"},{"key":"S0022215118001329_ref21","doi-asserted-by":"publisher","DOI":"10.1288\/00005537-198007000-00018"},{"key":"S0022215118001329_ref2","doi-asserted-by":"publisher","DOI":"10.1007\/BF02158983"},{"key":"S0022215118001329_ref23","doi-asserted-by":"publisher","DOI":"10.1002\/lary.1983.93.4.429"},{"key":"S0022215118001329_ref20","doi-asserted-by":"publisher","DOI":"10.1177\/000348948209100406"},{"key":"S0022215118001329_ref15","doi-asserted-by":"publisher","DOI":"10.1111\/ijd.1978.17.5.388"},{"key":"S0022215118001329_ref11","first-page":"43","article-title":"Predominantly cerebral manifestation in Urbach-Wiethe's syndrome (lipoid proteinosis cutis et mucosae): a clinical and pathomorphological study","volume":"6","author":"Kleinert","year":"1987","journal-title":"Clin Neuropathol"},{"key":"S0022215118001329_ref6","first-page":"1","article-title":"Urbach-Wiethe disease (lipoglycoproteinosis; lipoid proteinosis; hyalinosis cutis et mucosae). A review","volume":"53","author":"Hofer","year":"1973","journal-title":"Acta Dermatol Venereol Suppl (Stockh)"},{"key":"S0022215118001329_ref4","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/11.7.833"},{"key":"S0022215118001329_ref18","first-page":"767","article-title":"Otolaryngological manifestations and genetic characteristics of lipoid proteinosis","volume":"119","author":"Xu","year":"2010","journal-title":"Ann Otol Rhinol Laryngol"},{"key":"S0022215118001329_ref16","doi-asserted-by":"crossref","first-page":"233","DOI":"10.4103\/0976-237X.156053","article-title":"Lipoid proteinosis: a review with two case reports","volume":"6","author":"Kabre","year":"2015","journal-title":"Contemp Clin Dent"},{"key":"S0022215118001329_ref19","first-page":"128","article-title":"A case report: hybrid treatment approach to lipoid proteinosis of the larynx","volume":"31","author":"Chen","year":"2015","journal-title":"J Voice"}],"container-title":["The Journal of Laryngology &amp; Otology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.cambridge.org\/core\/services\/aop-cambridge-core\/content\/view\/S0022215118001329","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2019,4,13]],"date-time":"2019-04-13T15:49:18Z","timestamp":1555170558000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.cambridge.org\/core\/product\/identifier\/S0022215118001329\/type\/journal_article"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2018,8,13]]},"references-count":24,"journal-issue":{"issue":"10","published-print":{"date-parts":[[2018,10]]}},"alternative-id":["S0022215118001329"],"URL":"https:\/\/doi.org\/10.1017\/s0022215118001329","relation":{},"ISSN":["0022-2151","1748-5460"],"issn-type":[{"value":"0022-2151","type":"print"},{"value":"1748-5460","type":"electronic"}],"subject":[],"published":{"date-parts":[[2018,8,13]]}}}