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Laryngeal cleft

MedGen UID:
327075
Concept ID:
C1840311
Congenital Abnormality; Finding
Synonyms: Anterior submucous laryngeal cleft (subtype); Cleft, larynx posterior; Congenital laryngeal clefts (subtype); Posterior laryngeal cleft (PLC); Type 1A (minor laryngeal cleft)
SNOMED CT: Congenital cleft larynx (232461002); Congenital fissure of larynx (232461002)
 
HPO: HP:0008751

Definition

Presence of a gap in the posterior laryngotracheal wall with a continuity between the larynx and the esophagus. [from HPO]

Conditions with this feature

Pallister-Hall syndrome
MedGen UID:
120514
Concept ID:
C0265220
Disease or Syndrome
GLI3-related Pallister-Hall syndrome (GLI3-PHS) is characterized by a spectrum of anomalies ranging from polydactyly, asymptomatic bifid epiglottis, and hypothalamic hamartoma at the mild end to laryngotracheal cleft with neonatal lethality at the severe end. Individuals with mild GLI3-PHS may be incorrectly diagnosed as having isolated postaxial polydactyly type A. Individuals with GLI3-PHS can have pituitary insufficiency and may die as neonates from undiagnosed and untreated adrenal insufficiency.
X-linked Opitz G/BBB syndrome
MedGen UID:
424842
Concept ID:
C2936904
Disease or Syndrome
X-linked Opitz G/BBB syndrome (X-OS) is a multiple-congenital-anomaly disorder characterized by facial anomalies (hypertelorism, prominent forehead, widow's peak, broad nasal bridge, anteverted nares), genitourinary abnormalities (hypospadias, cryptorchidism, and hypoplastic/bifid scrotum), and laryngotracheoesophageal defects. Developmental delay and intellectual disability are observed in about 50% of affected males. Cleft lip and/or palate are present in approximately 50% of affected individuals. Other malformations (present in <50% of individuals) include congenital heart defects, imperforate or ectopic anus, and midline brain defects (Dandy-Walker malformation and agenesis or hypoplasia of the corpus callosum and/or cerebellar vermis). Wide clinical variability occurs even among members of the same family. Female heterozygotes usually manifest hypertelorism only.
Auriculocondylar syndrome 3
MedGen UID:
816662
Concept ID:
C3810332
Disease or Syndrome
Auriculocondylar syndrome (ARCND) is a rare craniofacial disorder involving first and second pharyngeal arch derivatives and includes the key features of micrognathia, temporomandibular joint and condyle anomalies, microstomia, prominent cheeks, and question mark ears (QMEs). QMEs consist of a defect between the lobe and the upper two-thirds of the pinna, ranging from a mild indentation in the helix to a complete cleft between the lobe and helix (summary by Gordon et al., 2013). For a general phenotypic description and discussion of genetic heterogeneity of auriculocondylar syndrome, see ARCND1 (602483).
PHIP-related behavioral problems-intellectual disability-obesity-dysmorphic features syndrome
MedGen UID:
1641154
Concept ID:
C4693860
Disease or Syndrome
Chung-Jansen syndrome (CHUJANS) is characterized by global developmental delay apparent from infancy, impaired intellectual development or learning difficulties, behavioral abnormalities, dysmorphic features, and obesity. The severity of the phenotype and additional features are variable (summary by Jansen et al., 2018).
Oculogastrointestinal-neurodevelopmental syndrome
MedGen UID:
1779113
Concept ID:
C5543355
Disease or Syndrome
Oculogastrointestinal neurodevelopmental syndrome (OGIN) is characterized by microphthalmia and/or coloboma in association with other congenital anomalies, including imperforate anus, horseshoe kidney, and structural cardiac defects. Hearing loss and severe developmental delay are also observed in most patients (Zha et al., 2020; Mor-Shaked et al., 2021).
Developmental delay, impaired speech, and behavioral abnormalities
MedGen UID:
1794167
Concept ID:
C5561957
Disease or Syndrome
Developmental delay, impaired speech, and behavioral abnormalities (DDISBA) is characterized by global developmental delay apparent from early childhood. Intellectual disability can range from mild to severe. Additional variable features may include dysmorphic facial features, seizures, hypotonia, motor abnormalities such as Tourette syndrome or dystonia, and hearing loss (summary by Cousin et al., 2021).
Developmental delay, hypotonia, and impaired language
MedGen UID:
1823975
Concept ID:
C5774202
Disease or Syndrome
Developmental delay, hypotonia, and impaired language (DEDHIL) is a neurodevelopmental disorder characterized by variably impaired intellectual development usually with hypotonia, mild motor delay, and language difficulties. Affected individuals may also have nonspecific dysmorphic facial features, gastrointestinal problems, and abnormalities on brain imaging (Stephenson et al., 2022).

Professional guidelines

PubMed

Reddy P, Byun YJ, Downs J, Nguyen SA, White DR
Int J Pediatr Otorhinolaryngol 2020 Nov;138:110370. Epub 2020 Sep 9 doi: 10.1016/j.ijporl.2020.110370. PMID: 33152963
Bowe SN, Hartnick CJ
Curr Opin Otolaryngol Head Neck Surg 2017 Dec;25(6):506-513. doi: 10.1097/MOO.0000000000000414. PMID: 28914624
Johnston DR, Watters K, Ferrari LR, Rahbar R
Int J Pediatr Otorhinolaryngol 2014 Jun;78(6):905-11. Epub 2014 Mar 27 doi: 10.1016/j.ijporl.2014.03.015. PMID: 24735606

Recent clinical studies

Etiology

van Stigt MJB, van Oorschot HD, Bittermann AJN
BMJ Case Rep 2022 Mar 2;15(3) doi: 10.1136/bcr-2021-247014. PMID: 35236684Free PMC Article
Padia R, Coppess S, Horn DL, Parikh SR, Hoang J, Faherty A, DeMarre K, Johnson K
Laryngoscope 2019 Nov;129(11):2588-2593. Epub 2019 Jan 23 doi: 10.1002/lary.27535. PMID: 30671968
Landry AM, Rutter MJ
Clin Perinatol 2018 Dec;45(4):597-607. Epub 2018 Sep 11 doi: 10.1016/j.clp.2018.07.002. PMID: 30396407
Trachsel D, Hammer J
Pediatr Pulmonol 2018 Sep;53(9):1284-1287. Epub 2018 Jul 3 doi: 10.1002/ppul.24117. PMID: 29971952
Hamilton J, Yaneza MM, Clement WA, Kubba H
Int J Pediatr Otorhinolaryngol 2016 Feb;81:1-4. Epub 2015 Dec 8 doi: 10.1016/j.ijporl.2015.11.027. PMID: 26810279

Diagnosis

Keane A, Leeper LK, Drake AF
Otolaryngol Clin North Am 2024 Aug;57(4):559-568. Epub 2024 Mar 19 doi: 10.1016/j.otc.2024.02.014. PMID: 38503669
Landry AM, Rutter MJ
Clin Perinatol 2018 Dec;45(4):597-607. Epub 2018 Sep 11 doi: 10.1016/j.clp.2018.07.002. PMID: 30396407
Strychowsky JE, Rahbar R
Semin Pediatr Surg 2016 Jun;25(3):128-31. Epub 2016 Feb 21 doi: 10.1053/j.sempedsurg.2016.02.005. PMID: 27301597
Windsor A, Clemmens C, Jacobs IN
Paediatr Respir Rev 2016 Jan;17:24-8. Epub 2015 Jul 10 doi: 10.1016/j.prrv.2015.07.001. PMID: 26277452
Pezzettigotta SM, Leboulanger N, Roger G, Denoyelle F, Garabédian EN
Otolaryngol Clin North Am 2008 Oct;41(5):913-33, ix. doi: 10.1016/j.otc.2008.04.010. PMID: 18775342

Therapy

Arnold L, Kennel C, Gov-Ari E
Int J Pediatr Otorhinolaryngol 2020 Nov;138:110279. Epub 2020 Aug 5 doi: 10.1016/j.ijporl.2020.110279. PMID: 32810687
Enver N, Asya O, Abuzaid G, Gürol E
J Voice 2020 Sep;34(5):812.e5-812.e8. Epub 2019 Apr 17 doi: 10.1016/j.jvoice.2019.03.011. PMID: 31005447
Burton Wood C, Rieber AK, Penn EB
Laryngoscope 2019 Jul;129(7):1696-1698. Epub 2018 Nov 19 doi: 10.1002/lary.27306. PMID: 30450589
Watters K, Ferrari L, Rahbar R
Laryngoscope 2013 Jan;123(1):264-8. Epub 2012 Aug 2 doi: 10.1002/lary.23498. PMID: 22865688
Mangat HS, El-Hakim H
Otolaryngol Head Neck Surg 2012 May;146(5):764-8. Epub 2012 Jan 18 doi: 10.1177/0194599811434004. PMID: 22261489

Prognosis

Seidl E, Kramer J, Hoffmann F, Schön C, Griese M, Kappler M, Lisec K, Hubertus J, von Schweinitz D, Di Dio D, Sittel C, Reiter K
Pediatr Pulmonol 2021 Jan;56(1):138-144. Epub 2020 Nov 5 doi: 10.1002/ppul.25133. PMID: 33095514
Landry AM, Rutter MJ
Clin Perinatol 2018 Dec;45(4):597-607. Epub 2018 Sep 11 doi: 10.1016/j.clp.2018.07.002. PMID: 30396407
Sobol SE
Curr Probl Pediatr Adolesc Health Care 2018 Mar;48(3):82-84. doi: 10.1016/j.cppeds.2018.01.007. PMID: 29571545
Arslankoylu AE, Unal E, Kuyucu N, Ismi O
Acta Otorhinolaryngol Ital 2016 Oct;36(5):431-434. doi: 10.14639/0392-100X-636. PMID: 27070536Free PMC Article
Hughes CA, Miller KA, Holinger LD, Miller RP
Int J Pediatr Otorhinolaryngol 2000 Aug 31;54(2-3):153-8. doi: 10.1016/s0165-5876(00)00346-3. PMID: 10967387

Clinical prediction guides

Wineski RE, Panico E, Karas A, Rosen P, Van Diver B, Norwood TG, Grayson JW, Beltran-Ale G, Dimmitt R, Kassel R, Rogers A, Leonard M, Chapman A, Boehm L, Wiatrak B, Harris WT, Smith N
Int J Pediatr Otorhinolaryngol 2021 Nov;150:110874. Epub 2021 Aug 6 doi: 10.1016/j.ijporl.2021.110874. PMID: 34392101
Hseu A, Ayele N, Dombrowski N, Irace A, Kawai K, Woodnorth G, Watters K, Nuss R, Rahbar R
Int J Pediatr Otorhinolaryngol 2019 Mar;118:59-61. Epub 2018 Dec 16 doi: 10.1016/j.ijporl.2018.12.018. PMID: 30583194
Bruhat C, Briac T, Delabaere A, Labbé A, Lémery D, Laurichesse-Delmas H, Gallot D
J Gynecol Obstet Hum Reprod 2018 Dec;47(10):577-579. Epub 2018 Sep 16 doi: 10.1016/j.jogoh.2018.09.003. PMID: 30232040
Arslankoylu AE, Unal E, Kuyucu N, Ismi O
Acta Otorhinolaryngol Ital 2016 Oct;36(5):431-434. doi: 10.14639/0392-100X-636. PMID: 27070536Free PMC Article
Bershof JF, Guyuron B, Olsen MM
J Craniomaxillofac Surg 1992 Jan;20(1):24-7. doi: 10.1016/s1010-5182(05)80192-1. PMID: 1564117

Recent systematic reviews

Timashpolsky A, Schild SD, Ballard DP, Leventer SP, Rosenfeld RM, Plum AW
Otolaryngol Head Neck Surg 2021 Mar;164(3):489-500. Epub 2020 Aug 18 doi: 10.1177/0194599820947742. PMID: 32807006
Reddy P, Byun YJ, Downs J, Nguyen SA, White DR
Int J Pediatr Otorhinolaryngol 2020 Nov;138:110370. Epub 2020 Sep 9 doi: 10.1016/j.ijporl.2020.110370. PMID: 33152963

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