Laryngeal Cleft: Diagnosis and Treatment of an Unusual Presentation in an Adult

Garcia-Lopez, Isabel; Perez-Fernandez, Carmen Aranzazu; Garcia-Raya, Pilar; Gavilan, Javier

doi:10.1016/j.otoeng.2019.05.002

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(A) Axial CT: incomplete fusion of the cricoid cartilage (arrow). (B) Coronal CT: defect in the upper part of the cricoid cartilage (arrow)." ] ] ] "textoCompleto" => "Clinical CaseA 54-year old male presented with severe dysphonia of long duration with the impossibility of hearing anything in noisy ambiances. He did not spontaneously mention any changes in swallowing function although when specifically asked about this he referred to having always gagged on swallowing liquids. He had not had pneumonia. Vocal incapacity rate (VHI-10) was 18/40.Perceptual assessment with the GRBAS index was G3R1B3A1S3. Aerodynamic assessment showed maximum phonation time (MPT) of 2s. Maximum intensity was 97dB.Laryngeal stroboscopy showed a striking defect of posterior closure, large in size, with normal mobility of both vocal cords (adduction and abduction). The presence of redundant tissue in the interarytenoid space was observed (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>).<elsevierMultimedia ident="fig0005"></elsevierMultimedia>Computerised tomography (CT) showed an incomplete fusion between the upper parts that caused a defect of the cricoid cartilage layer (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>).<elsevierMultimedia ident="fig0010"></elsevierMultimedia>Surgical examination was performed under general anaesthesia confirming the presence of a type 2 laryngeal cleft.Following meticulous assessment of voice improvement expectations, the patient decided on surgical treatment.An endoscopic approach with extirpation of redundant mucous with CO2 laser was performed, suturing the remaining mucous with Vycril® 4-0. No postoperative complications ensued.VHI-10 improved up to a value of 12/40.MPT increased to 5s and postoperative GRBAS was G2R0B1A0S2.Pre and postoperative laryngeal endoscopic images are shown in <a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>.DiscussionLaryngeal clefts are usually diagnosed during the first few months or years of life. They are defined by the existence of a congenital communication between the hypopharynx and the larynx, which may be extended so as to communicate the trachea with the oesophagus.The most commonly used classification is that of Benjamin and Inglis,<a class="elsevierStyleCrossRef" href="#bib0030">1</a> which divides the laryngeal clefts into 4 types:<ul class="elsevierStyleList" id="lis0005"><li class="elsevierStyleListItem" id="lsti0005">–Type 1: supraglottic cleft</li><li class="elsevierStyleListItem" id="lsti0010">–Type 2: the cleft extends below the level of the vocal cords and involves part of the cricoid cartilage</li><li class="elsevierStyleListItem" id="lsti0015">–Type 3: the cleft extents through the whole of the cricoid cartilage and part of the cervical trachea</li><li class="elsevierStyleListItem" id="lsti0020">–Type 4: the cleft extends to the intra-thoracic trachea</li></ul>Main symptoms include dyspnoea, cough, gagging episodes, stridor and recurrent aspirations with repeated respiratory infections.In general the severity of symptoms is correlated with the magnitude of the cleft. Type 1 clefts are sometimes asymptomatic, whilst type 2 or larger ones usually present severe symptoms from birth.Diagnosis of laryngeal clefts may sometimes present a challenge to the ENT specialist. Diagnosis requires a high level of suspicion.The symptoms which usually present are related to swallowing, from difficulties with feeding during the first few weeks of life to the presence of aspiration pnemonias.<a class="elsevierStyleCrossRefs" href="#bib0035">2,3</a>It is extremely rare to diagnose this condition in adult age. As far as we are aware there are only 2 cases in the literature of patients older than ours when diagnosed.<a class="elsevierStyleCrossRefs" href="#bib0035">2,4,5</a>Furthermore, the fact that dysphonia was the essential symptom of the patient makes this case particularly unusual.On occasions laryngeal endoscopy does not clearly demonstrate the existence of a cleft. There are some indirect symptoms, such as those described by Weissbrod et al.<a class="elsevierStyleCrossRef" href="#bib0035">2</a> called “the ram sign”, which refers to the presence of redundant mucosa that has prolapsed between the arytenoids and the endoscopic image of which is similar to the horns of a ram. In our case this redundancy of interarytenoid mucous also existed with the presence of this sign (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>).Direct laryngoscopy under general anaesthesia with palpation of the interarytenoid region confirmed diagnosis. In our case, both laryngeal endoscopy and CT were performed prior to surgical examination so that an accurate diagnosis could be performed.Regarding treatment, surgical closure of the cleft usually improves deglutition. As far as we are aware, there are no publications regarding the improvement in vocal parameters after laryngeal cleft surgery, as shown in this case.Conflict of InterestsThe authors have no conflict of interests to declare" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Clinical Case" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Discussion" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Conflict of Interests" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2019-02-13" "fechaAceptado" => "2019-05-03" "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "Please cite this article as: Garcia-Lopez I, Perez-Fernandez CA, Garcia-Raya P, Gavilan J. Hendidura laríngea: diagnóstico y tratamiento en un caso inusual de presentación en el adulto. Acta Otorrinolaringol Esp. 2020;71:122–124." ] ] "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 714 "Ancho" => 1305 "Tamanyo" => 199899 ] ] "descripcion" => array:1 [ "en" => "Laryngeal endoscopy. (A) Preoperative. Defect of triangular (cleft) closure in the retrocricoid regions in adduction positions. The ram sign. (B) Postoperative. Closure of the posterior defect after surgery." ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 775 "Ancho" => 1255 "Tamanyo" => 55107 ] ] "descripcion" => array:1 [ "en" => "CT of the larynx. (A) Axial CT: incomplete fusion of the cricoid cartilage (arrow). (B) Coronal CT: defect in the upper part of the cricoid cartilage (arrow)." ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0030" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Minor congenital laryngeal clefts: diagnosis and classification" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "B. Benjamin" 1 => "A. 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Merati" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1177/000348941212100807" "Revista" => array:6 [ "tituloSerie" => "Ann Otol Rhinol Laryngol" "fecha" => "2012" "volumen" => "121" "paginaInicial" => "533" "paginaFinal" => "538" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/22953660" "web" => "Medline" ] ] ] ] ] ] ] ] 2 => array:3 [ "identificador" => "bib0040" "etiqueta" => "3" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Minimally invasive approach to laryngeal cleft" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "K. Watters" 1 => "L. Ferrari" 2 => "R. 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Sandhu" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1308/003588414X14055925059156" "Revista" => array:6 [ "tituloSerie" => "Ann R Coll Surg Engl" "fecha" => "2015" "volumen" => "97" "paginaInicial" => "e6" "paginaFinal" => "e8" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/25519258" "web" => "Medline" ] ] ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0050" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "A case of adult congenital laryngeal cleft asymptomatic until hypopharynx cancer treatment" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "K. Shimizu" 1 => "A. Uno" 2 => "K. Takemura" 3 => "N. Ashida" 4 => "R. Oya" 5 => "T. Kitamura" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.anl.2017.09.004" "Revista" => array:6 [ "tituloSerie" => "Auris Nasus Larynx" "fecha" => "2018" "volumen" => "45" "paginaInicial" => "640" "paginaFinal" => "643" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/28943051" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/21735735/0000007100000002/v1_202004040728/S2173573520300302/v1_202004040728/en/main.assets" "Apartado" => array:4 [ "identificador" => "5881" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Case Studies" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/21735735/0000007100000002/v1_202004040728/S2173573520300302/v1_202004040728/en/main.pdf?idApp=UINPBA00004N&text.app=https://www.elsevier.es/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2173573520300302?idApp=UINPBA00004N"]

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Acta Otorrinolaringológica Española, the official publication of the Spanish Society of Otolaryngology and Head and Neck Surgery and the Ibero-American Society of Otorhinolaryngology, is the most important journal in Spanish dedicated to Otorhinolaryngology, which reflects the continuous scientific and technical progress of the specialty. It has become an important tool to be kept up to date. The Journal publishes Original Articles (articles on basic research and clinical investigation), reviews, brief communications, case studies and images, which are subjected to a rigorous peer review process.

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Impact factor

The Impact Factor measures the average number of citations received in a particular year by papers published in the journal during the two preceding years.

© Clarivate Analytics, Journal Citation Reports 2022

Impact factor 2023

0.9

Citescore

CiteScore measures average citations received per document published.

Citescore 2023

1.4

SJR

SRJ is a prestige metric based on the idea that not all citations are the same. SJR uses a similar algorithm as the Google page rank; it provides a quantitative and qualitative measure of the journal's impact.

SJR 2023

0.326

SNIP

SNIP measures contextual citation impact by wighting citations based on the total number of citations in a subject field.

SNIP 2023

0.54

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Journal Information

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Vol. 71. Issue 2.