|
1
|
Maldonado A, Muñoz R, Cabrera J, de Arruda JAA, de Andrade BAB, Villarroel-Dorrego M, do Valle IB. Heterotopic gastrointestinal cyst of the oral cavity: A rare clinical report and literature review. JOURNAL OF STOMATOLOGY, ORAL AND MAXILLOFACIAL SURGERY 2025:102406. [PMID: 40350078 DOI: 10.1016/j.jormas.2025.102406] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Subscribe] [Scholar Register] [Received: 01/23/2025] [Revised: 03/31/2025] [Accepted: 05/08/2025] [Indexed: 05/14/2025]
Abstract
This study reports a case of oral heterotopic gastrointestinal cyst (HGIC) and provides a comprehensive analysis of its demographic distribution, clinicopathological features, and treatment outcomes based on a literature review. The case involved a 6-year-old boy with a sublingual HGIC, with detailed documentation of the clinical presentation, imaging findings, surgical management, and histopathological features. Searches were conducted in PubMed, Scopus, Web of Science, Embase, and LILACS, supplemented by manual searches/gray literature. The patient exhibited a well-defined sublingual tumor, which was successfully treated by surgical excision. Histopathological analysis revealed both intestinal and gastric epithelium lining the cyst. The searches identified 111 cases of oral HGIC. The mean patient age was 5.4 years, with a slight male predominance. The tongue was the most frequently affected site, followed by the floor of the mouth. Larger cysts were associated with airway obstruction, feeding difficulties, or speech impairment. Microscopically, the predominant epithelial components were gastric, intestinal, and squamous. Surgical excision was the primary treatment and demonstrated low recurrence rates. Although rare, oral HGIC requires a high index of clinical suspicion due to its potential to mimic common oral lesions. Recognition of the diverse epithelium components is crucial for improving diagnostic accuracy.
Collapse
Affiliation(s)
- Andrea Maldonado
- Postgrado de Cirugía Bucomaxilofacial, Universidad de Carabobo, Valencia, Venezuela
| | - Rubén Muñoz
- Postgrado de Cirugía Bucomaxilofacial, Universidad de Carabobo, Valencia, Venezuela
| | - Jesús Cabrera
- Postgrado de Cirugía Bucomaxilofacial, Universidad de Carabobo, Valencia, Venezuela
| | - José Alcides Almeida de Arruda
- Department of Oral Diagnosis and Pathology, School of Dentistry, Universidade Federal do Rio de Janeiro, Rio de Janeiro, Brazil.
| | | | | | | |
Collapse
|
|
2
|
Aforka EE, Salami AS, Okhuaihesuyi O, Akinshipo AW, Afolabi JB. Lingual Cyst with Respiratory Epithelium: An Update of Scientific Literature. Niger Postgrad Med J 2025; 32:160-164. [PMID: 40364686 DOI: 10.4103/npmj.npmj_20_25] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/25/2025] [Accepted: 03/29/2025] [Indexed: 05/15/2025]
Abstract
Tongue (lingual) cysts occurring on the dorsal or ventral surfaces present as swellings. These swellings could cause difficulty in eating, drinking, speaking or breathing. Lingual cyst with respiratory epithelium (LCRE) is a rare developmental cyst of which about 23 case reports exist in the literature. This report aimed to document a case of LCRE from our centre to update the literature and to distinguish it from other cysts of the tongue with or without respiratory epithelium in addition to other content. We present a case of lingual cyst in a 4-year-old boy of 4 years; histopathologic features revealed a cystic lesion lined by columnar ciliated epithelium with goblet cells, focal areas of squamous epithelium and areas of inflammation in the connective tissue. This confirms a diagnosis of LCRE. A discussion on the clinical and microscopic features that distinguishes it from the other tongue cyst is presented, and the need for an appropriate nomenclature is highlighted.
Collapse
Affiliation(s)
- Ernest Ebuka Aforka
- Department of Oral Pathology and Oral Medicine, University of Nigeria Teaching Hospital, Ituku/Ozalla, Enugu State, Nigeria
| | - Afolake Shakirat Salami
- Department of Oral/Maxillofacial Pathology, Radiology and Oral Medicine, University of Medical Sciences, Ondo, Nigeria
| | - Osariemen Okhuaihesuyi
- Department of Integrated Human Sciences, Missouri School of Dentistry and Oral Health (MOSDOH), A T Still University, Kirksville, MO, USA
| | - Abdul-Warith Akinshipo
- Department of Oral and Maxillofacial Pathology, College of Medicine, University of Lagos, Idi-Araba, Lagos, Nigeria
| | - Jacob Babatunde Afolabi
- Department of Child Dental Health, Obafemi Awolowo University Teaching Hospital Complex, Ile Ife, Osun State, Nigeria
| |
Collapse
|
|
3
|
Ma B, Zeng Q, Le J, Wicker J, Powell K. Lingual Bronchogenic Cyst: A Case Report and Literature Review of a Rare Pathology. Cureus 2025; 17:e78451. [PMID: 40051932 PMCID: PMC11882358 DOI: 10.7759/cureus.78451] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 02/03/2025] [Indexed: 03/09/2025] Open
Abstract
Choristomas are rare, benign embryonic tumors characterized by normal tissue appearing in abnormal sites. When lined with respiratory epithelium, they are classified as bronchogenic cysts. While these cysts most commonly occur in the mediastinum or other thoracic regions, their occurrence on the tongue is exceedingly rare. Here, we present the case of a four-year-old boy diagnosed with a bronchogenic cyst of the tongue, highlighting its clinical and histological features, diagnosis, and management, along with a review of the literature.
Collapse
Affiliation(s)
- Boyu Ma
- Oral and Maxillofacial Surgery, University of Alabama at Birmingham, Birmingham, USA
| | - Qingcong Zeng
- Oral and Maxillofacial Surgery, University of Alabama at Birmingham, Birmingham, USA
| | - John Le
- Oral and Maxillofacial Surgery, University of Florida College of Medicine-Jacksonville, Jacksonville, USA
| | - James Wicker
- Anatomic Pathology, University of Alabama at Birmingham, Birmingham, USA
| | - Kathlyn Powell
- Oral and Maxillofacial Surgery, University of Alabama at Birmingham, Birmingham, USA
| |
Collapse
|
|
4
|
Varlas VN, Parlatescu I, Epistatu D, Neagu O, Varlas RG, Bălănescu L. Mixed Heterotopic Gastrointestinal/Respiratory Oral Cysts in Newborns: From Prenatal Diagnosis to Histopathological and Therapeutic Management: A Case Report and Literature Review. Diagnostics (Basel) 2024; 14:339. [PMID: 38337855 PMCID: PMC10855558 DOI: 10.3390/diagnostics14030339] [Citation(s) in RCA: 2] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/09/2023] [Revised: 01/21/2024] [Accepted: 01/31/2024] [Indexed: 02/12/2024] Open
Abstract
Fetal lingual tumors are very rare, and their early prenatal diagnosis is important for defining the subsequent therapeutic strategy. In this study, we aimed to describe a case of a congenital septate lingual cyst and perform an extensive literature review on two main databases (PubMed, Web of Science), analyzing the clinical manifestations, the imaging appearance, the differential diagnosis, and particularities regarding the treatment of these tumors. The electronic search revealed 17 articles with 18 cases of mixed heterotopic gastrointestinal/respiratory oral epithelial cysts that met the eligibility criteria and were included in this review. The clinical case was diagnosed prenatally during second-trimester screening. On the eighth day of life, the fetus underwent an MRI of the head, which revealed an expansive cystic process on the ventral side of the tongue with the greatest diameter of 21.7 mm, containing a septum of 1 mm inside. On the 13th day of life, surgery was performed under general anesthesia, and the lingual cystic formation was completely excised. The postoperative evolution was favorable. The histopathological examination revealed a heterotopic gastric/respiratory-mixed epithelial cyst with non-keratinized respiratory, gastric squamous, and foveolar epithelium. The lingual cyst diagnosed prenatally is an accidental discovery, the differential diagnosis of which can include several pathologies with different degrees of severity but with a generally good prognosis.
Collapse
Affiliation(s)
- Valentin Nicolae Varlas
- Department of Obstetrics and Gynecology, Filantropia Clinical Hospital, 011132 Bucharest, Romania; (V.N.V.); (R.G.V.)
- Faculty of Dentistry, Carol Davila University of Medicine and Pharmacy, 010221 Bucharest, Romania
| | - Ioanina Parlatescu
- Department of Oral Pathology, Faculty of Dentistry, Carol Davila University of Medicine and Pharmacy, 17-21, Calea Plevnei Street, 020021 Bucharest, Romania
| | - Dragos Epistatu
- Department of Radiology, Faculty of Dentistry, Carol Davila University of Medicine and Pharmacy, 17-21, Calea Plevnei Street, 020021 Bucharest, Romania
| | - Oana Neagu
- Department of Anatomopathology, Carol Davila University of Medicine and Pharmacy, 050474 Bucharest, Romania;
| | - Roxana Georgiana Varlas
- Department of Obstetrics and Gynecology, Filantropia Clinical Hospital, 011132 Bucharest, Romania; (V.N.V.); (R.G.V.)
| | - Laura Bălănescu
- Department of Pediatric Surgery, Children Emergency Hospital “Grigore Alexandrescu”, 011743 Bucharest, Romania;
- Faculty of General Medicine, Carol Davila University of Medicine and Pharmacy, 050474 Bucharest, Romania
| |
Collapse
|
|
5
|
Philipone E, Yoon AJ. Mucosal Soft Tissue Lesions. Pediatr Dev Pathol 2023; 26:596-608. [PMID: 37974086 DOI: 10.1177/10935266231198724] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/19/2023]
Abstract
Mucosal soft tissue lesions are fairly common in the pediatric population. However, the precise prevalence is unknown. This is the result of the limited number of studies, the use of various diagnostic criteria in those studies, and the transient nature of commonly encountered lesions in this population. In this section, we seek to familiarize the pediatric pathologist with a sampling of mucosal soft tissue lesions encountered in pediatric patients, highlight key diagnostic features and correlations with systemic diseases should they exist.
Collapse
Affiliation(s)
- Elizabeth Philipone
- Division of Oral and Maxillofacial Pathology, Department of Pathology and Cell Biology, Columbia University Irving Medical Center, New York, NY, USA
| | - Angela J Yoon
- Medical University of South Carolina, Charleston, SC, USA
| |
Collapse
|
|
6
|
Cialente F, De Soccio G, Savastano V, Grasso M, Dello Spedale Venti M, Ralli M, Riminucci M, De Vincentiis M, Corsi A, Antonio Minni. Lingual cyst with respiratory epithelium: The importance of differential diagnosis. Bosn J Basic Med Sci 2021; 21:378-382. [PMID: 32464085 PMCID: PMC8112560 DOI: 10.17305/bjbms.2020.4716] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/28/2020] [Accepted: 05/20/2020] [Indexed: 11/16/2022] Open
Abstract
Lingual cyst with respiratory epithelium (LCRE) is a very rare congenital cyst of the tongue, floor of the mouth, pharynx or hypopharynx with 21 cases reported in the literature (1, 2). Differential diagnosis is very important for patients presenting with lingual cysts, as this may impact treatment and follow-up. LCRE should be included in the different diagnosis of dermoid cyst (3), teratoid cyst (4), epidermoid cyst (5), thyroglossal duct cyst (6), lymphoepithelial cyst (7), mucocele or ranula (8). Each entity has a peculiar histologic presentation, although the clinical aspect may be very similar (1). The dermoid cyst is lined by a keratinized squamous epithelium and contains skin appendages in the cyst. Epidermoid cyst is similar to the dermoid cyst but is characterized by non-keratinized squamous epithelium and has a lumen filled of keratin. Teratoid cyst contains derivatives of the endoderm, ectoderm and/or mesoderm. The thyroglossal duct cyst is usually lined by columnar, stratified squamous epithelium, or an intermediate transition type of epithelium, with the mandatory presence of thyroid tissue in the cyst wall. Lymphoepithelial cyst is identified by the presence of the lymphoid aggregates in the cyst wall. A mucous retention cyst, so called mucocele or ranula, contains mucin and granulation tissue (1).
Read the full text [PDF]
Collapse
Affiliation(s)
- Fabrizio Cialente
- Department of Sense Organs, University Sapienza of Rome, Rome, Italy
| | - Giulia De Soccio
- Department of Sense Organs, University Sapienza of Rome, Rome, Italy
| | - Vincenzo Savastano
- UOSD Pediatric ENT, DAI Head-Neck, University Hospital Policlinico Umberto I, Rome, Italy
| | - Michele Grasso
- Department of Sense Organs, University Sapienza of Rome, Rome, Italy
| | | | - Massimo Ralli
- Department of Sense Organs, University Sapienza of Rome, Rome, Italy
| | - Mara Riminucci
- Department of Molecular Medicine, University Sapienza of Rome, Rome, Italy
| | - Marco De Vincentiis
- Department of Oral and Maxillo-Facial Surgery, University Sapienza of Rome, Rome, Italy
| | - Alessandro Corsi
- Department of Molecular Medicine, University Sapienza of Rome, Rome, Italy
| | - Antonio Minni
- Department of Sense Organs, University Sapienza of Rome, Rome, Italy
| |
Collapse
|
|
7
|
Bilodeau EA, Hunter KD. Odontogenic and Developmental Oral Lesions in Pediatric Patients. Head Neck Pathol 2021; 15:71-84. [PMID: 33723756 PMCID: PMC8010029 DOI: 10.1007/s12105-020-01284-3] [Citation(s) in RCA: 11] [Impact Index Per Article: 2.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/23/2020] [Accepted: 12/31/2020] [Indexed: 11/29/2022]
Abstract
This article reviews odontogenic and developmental oral lesions encountered in the gnathic region of pediatric patients. The process of odontogenesis is discussed as it is essential to understanding the pathogenesis of odontogenic tumors. The clinical presentation, microscopic features, and prognosis are addressed for odontogenic lesions in the neonate (dental lamina cysts/gingival cysts of the newborn, congenital (granular cell) epulis of the newborn, melanotic neuroectodermal tumor, choristoma/heterotopia, cysts of foregut origin), lesions associated with unerupted/erupting teeth (hyperplastic dental follicle, eruption cyst, dentigerous cyst, odontogenic keratocyst/keratocystic odonogenic tumor, buccal bifurcation cyst/inflammatory collateral cyst) and pediatric odontogenic hamartomas and tumors (odontoma, ameloblastic fibroma, ameloblastoma, adenomatoid odontogenic tumor, primordial odontogenic tumor). Pediatric odontogenic and developmental oral lesions range from common to rare, but familiarity with these entities is essential due to the varying management implications of these diagnoses.
Collapse
Affiliation(s)
- Elizabeth A Bilodeau
- School of Dental Medicine, University of Pittsburgh, G-135 Salk Hall, 3501 Terrace Street, Pittsburgh, PA 15261 USA
| | - Keith D Hunter
- Academic Unit of Oral and Maxillofacial Medicine and Pathology, School of Clinical Dentistry, University of Sheffield, Sheffield, UK
| |
Collapse
|
|
8
|
Abstract
Bronchogenic cysts are foregut-derived developmental anomalies found along the developmental pathway of the foregut. The putative theory of pathogenesis is abnormal budding or branching of epithelial cells during the development of tracheobronchial tree. Over 99 % of cases occur in the mediastinum and lung while the head and neck area is affected in less than 1 % of cases with only rare cases reported in the oral cavity. This is a report of a case of a bronchogenic cyst arising in a 6-year-old male. The lesion presented as a painless swelling of the left underside of the tongue. Microscopically, the cyst was lined by pseudostratified columnar epithelium exhibiting many ciliated and mucous cells. A focus of cartilage and discontinuous bundles of smooth muscle were present adjacent to the lining. Where there was cyst rupture, there was granulation tissue associated with many foamy macrophages and acute and chronic inflammation. Four other cases, three in the tongue and one in the lower lip vestibule with cutaneous extension, all in the midline, have been reported in a 1 day-old male, 4 year-old male, 6 year-old female and 3 year-old male. There was no recurrence after excision and this is in keeping with the behavior in previous reports. Other developmental cysts including foregut cysts may be focally lined with respiratory epithelium but the presence of cartilage is the sine qua non for the diagnosis of a bronchogenic cyst.
Collapse
|
|
9
|
Lee AD, Harada K, Tanaka S, Yokota Y, Mima T, Enomoto A, Kogo M. Large lingual heterotopic gastrointestinal cyst in a newborn: A case report. World J Clin Cases 2020; 8:3808-3813. [PMID: 32953857 PMCID: PMC7479572 DOI: 10.12998/wjcc.v8.i17.3808] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/26/2020] [Revised: 08/01/2020] [Accepted: 08/19/2020] [Indexed: 02/05/2023] Open
Abstract
BACKGROUND Heterotopic gastrointestinal cysts have gastrointestinal epithelium in the cyst wall and rarely occur in the oral cavity. Most are found in the neonatal period. However, heterotopic gastrointestinal cysts that are diagnosed as a congenital tongue cyst by routine ultrasonography are extremely rare.
CASE SUMMARY A 12-day-old female presented with swelling of the anterior tongue. The obstetrician had detected significant tongue swelling on fetal ultrasonography in the 35th gestational week. The female was born by cesarean delivery at gestational week 39. She soon became dyspneic, and the cyst was aspirated. After the aspiration, her breathing recovered and she started breastfeeding. The cyst was excised under general anesthesia on the 67th day. Histopathologic examination showed that that cyst wall consisted of a lining of columnar gastrointestinal-type epithelium and pseudostratified ciliated epithelium. The patient restarted breastfeeding 3 h after surgery. The postoperative course was uneventful.
CONCLUSION Airway distress and feeding difficulty were successfully avoided by cyst aspiration, and surgical resection was performed with no perioperative complications.
Collapse
Affiliation(s)
- Atsushi-Doksa Lee
- First Department of Oral and Maxillofacial Surgery, Graduate School of Dentistry, Osaka University, Suita 565-0871, Osaka, Japan
- Department of Oral and Maxillofacial Surgery, Faculty of Medicine, Kindai University, Osaka-Sayama 589-8511, Osaka, Japan
| | - Kazuma Harada
- First Department of Oral and Maxillofacial Surgery, Graduate School of Dentistry, Osaka University, Suita 565-0871, Osaka, Japan
| | - Susumu Tanaka
- First Department of Oral and Maxillofacial Surgery, Graduate School of Dentistry, Osaka University, Suita 565-0871, Osaka, Japan
| | - Yusuke Yokota
- First Department of Oral and Maxillofacial Surgery, Graduate School of Dentistry, Osaka University, Suita 565-0871, Osaka, Japan
| | - Takashi Mima
- Department of Oral and Maxillofacial Surgery, Daini Osaka Police Hospital, Osaka 543-8922, Osaka, Japan
| | - Akifumi Enomoto
- Department of Oral and Maxillofacial Surgery, Faculty of Medicine, Kindai University, Osaka-Sayama 589-8511, Osaka, Japan
| | - Mikihiko Kogo
- First Department of Oral and Maxillofacial Surgery, Graduate School of Dentistry, Osaka University, Suita 565-0871, Osaka, Japan
| |
Collapse
|
|
10
|
Ardila CM, Álvarez-Martínez E. A Case Report of a Lingual Cyst Lined by Respiratory Epithelium in a Child. CLINICAL MEDICINE INSIGHTS-CASE REPORTS 2020; 13:1179547620919695. [PMID: 32528226 PMCID: PMC7263107 DOI: 10.1177/1179547620919695] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/05/2020] [Accepted: 03/26/2020] [Indexed: 11/16/2022]
Abstract
The lingual cyst lined by respiratory epithelium is a rare pathology. It probably appears from the default of undifferentiated cells of the foregut during embryonic growth. This pathology is seen more often in males and children; however, only 5 patients younger than 4 years old have been reported. The pathophysiology and the management of this cyst were described in a 4-year-old girl. She presented with a soft mass on the dorsum of the tongue covered by normal mucosa, which existed since her birth, causing difficulty in eating, breathing, and talking. The magnetic resonance described a hyperintense image with an anteroposterior diameter of 27 mm, craniocaudal of 19 mm, and transversal of 26 mm in the midline groove of the tongue; the scintigraphy showed normality. The enucleation of the lesion was performed, eradicating the capsule of the cyst and obtaining a complete cleavage. The histopathologic examination defined a cyst lined predominantly by respiratory epithelium. Unlike in other cases, in this case their cystic lining and capsular constituents were contemplated considering the current histological recommendations. It is relevant to differentiate this pathology from other cysts with similar histological findings.
Collapse
Affiliation(s)
- Carlos-Martín Ardila
- Biomedical Stomatology Research Group, Universidad de Antioquia, Medellín, Colombia
- Basic Sciences Department, Faculty of Dentistry, Universidad de Antioquia, Medellín, Colombia
| | - Efraín Álvarez-Martínez
- Basic Sciences Department, Faculty of Dentistry, Universidad de Antioquia, Medellín, Colombia
| |
Collapse
|
|
11
|
Diom ES, Fagan JJ, Bolding E. Intralingual Mucous Extravasation Cyst: An Uncommon Lingual Cyst. EAR, NOSE & THROAT JOURNAL 2019; 98:E21-E23. [PMID: 30943808 DOI: 10.1177/0145561319836303] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/16/2022] Open
Abstract
OBJECTIVE We report a rare case of an intralingual ranula. The differential diagnosis, etiology, diagnostic features, and management are discussed. CASE REPORT An 18-year-old man presented with a mass that extended along the ventral surface of the tongue and up to the tip. The computed tomography scan clearly defined the extent of a cystic lesion. The pathologic diagnosis of an intralingual ranula was made. CONCLUSION Lingual cysts have a varied etiology. Diagnosis hinges on histological examination of the cyst wall. Conservative resection and histological examination is the standard of care.
Collapse
Affiliation(s)
- Evelyne S Diom
- 1 Division of Otolaryngology, Groote Schuur Hospital, Cape Town, South Africa
| | - Johannes J Fagan
- 2 University of Cape Town Medical School, Cape Town, South Africa
| | - Ellen Bolding
- 3 Histopathologist, Pathcare, Cape Town, South Africa
| |
Collapse
|
|
12
|
Gunnarsdottir AB, Nilsson JS. Lingual bronchogenic cyst in a young child: A case report. ACTA OTO-LARYNGOLOGICA CASE REPORTS 2018. [DOI: 10.1080/23772484.2018.1486196] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/28/2022] Open
Affiliation(s)
- Agnes B. Gunnarsdottir
- Department of Otorhinolaryngology and Head and Neck Surgery, Skane University Hospital, Lund, Sweden
- Department of Clinical Sciences, Lund University, Lund, Sweden
| | - Johan S. Nilsson
- Department of Otorhinolaryngology and Head and Neck Surgery, Skane University Hospital, Lund, Sweden
- Department of Clinical Sciences, Lund University, Lund, Sweden
| |
Collapse
|
|
13
|
Peters SM, Park M, Perrino MA, Cohen MD. Lingual cyst with respiratory epithelium: report of 2 cases and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol 2018; 126:e279-e284. [PMID: 29929866 DOI: 10.1016/j.oooo.2018.05.011] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/26/2018] [Revised: 05/26/2018] [Accepted: 05/27/2018] [Indexed: 12/26/2022]
Abstract
The lingual cyst with respiratory epithelium, a congenital cyst of the tongue or floor of the mouth, is lined predominately by respiratory-type epithelium. The terminology for this lesion was first proposed in 1999 by Manor et al., who stated that a descriptive term is best for this cyst of debatable pathogenesis. Although it is a cyst of foregut origin, the lingual cyst with respiratory epithelium is distinguished from the traditional enteric (foregut) duplication cyst in that the latter contains gastric and/or intestinal-type mucosa and has smooth muscle within the cyst wall. This article presents 2 new cases of this entity, as well as reviews the 19 cases that have been reported in the literature and were found to fulfill the histologic criteria of an lingual cyst with respiratory epithelium.
Collapse
Affiliation(s)
- Scott M Peters
- Division of Oral and Maxillofacial Pathology, Columbia University Medical Center, New York, NY, USA
| | - Mark Park
- Department of Oral and Maxillofacial Surgery, Columbia University Medical Center, New York, NY, USA
| | - Michael A Perrino
- Department of Oral and Maxillofacial Surgery, Columbia University Medical Center, New York, NY, USA
| | - Molly D Cohen
- Division of Oral and Maxillofacial Pathology, The Mount Sinai Hospital, New York, NY, USA.
| |
Collapse
|
|
14
|
Martínez-Mata G, Inda-Cunningham D, Medina-López JA, Pacheco-Santiesteban R, Monreal-Romero H. Congenital lingual cyst: Expression of MUC protein and report of a new case. JOURNAL OF STOMATOLOGY, ORAL AND MAXILLOFACIAL SURGERY 2017; 118:376-378. [PMID: 28710001 DOI: 10.1016/j.jormas.2017.07.001] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 09/22/2016] [Revised: 06/27/2017] [Accepted: 07/05/2017] [Indexed: 11/30/2022]
Abstract
Lingual congenital cysts are uncommon lesions that alter the functions of speech, swallowing and breathing when they have considerable dimension. They usually appear from birth and increase in size gradually in childhood and adolescence. While there are a considerable number of case reports, the nomenclature and origin of this lesion are controversial. Congenital lingual cysts are composed of an epithelial lining that can show heterogeneous histological features, such as globed, ciliated, squamous and parietal cells, while the wall presents mature connective tissue and eventually smooth muscle. In the present manuscript, we report a case of a congenital lingual cyst in a 13-year-old boy, as well as the immunoexpression of MUC family proteins (MUC-1 and MUC-5AC), hoping to provide data that will help to clarify the possible etiology of this lesion.
Collapse
Affiliation(s)
- G Martínez-Mata
- Faculty of Medicine, University of Chihuahua, Chihuahua, Mexico.
| | | | | | | | | |
Collapse
|
|
15
|
Ramanathan M, Balasundharam S, Christabel A, Murali P, Pandem S. Simultaneous Occurrence of a Midline Sublingual Dermoid Cyst with Respiratory Epithelium and Submental Dermoid Cyst in a Paediatric Patient: A Case Report and Review of Literature. J Maxillofac Oral Surg 2016; 17:188-192. [PMID: 29618884 DOI: 10.1007/s12663-016-0972-9] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/05/2016] [Accepted: 09/27/2016] [Indexed: 12/22/2022] Open
Abstract
Aim To present the case of a simultaneously occuring sublingual dermoid cyst with respiratory epthelium and a submental dermoid cyst, and also to review literatures on the same. Methods A complete examination, radiograph, MRI and excision biopsy of both lesions were carried out and diagnosis was arrived at histopathologically. Results The histopathology of the submental swelling revealed orthokeratinized stratified squamous epithelium with underlying connective tissue consisting of dense irregularly arranged collagen fibres with fibroblasts along with chronic inflammatory cell infiltrate of lymphocytes and plasma cells. There was presence of sebaceous glands and sebum. Sublingual swelling showed non-keratinized stratified squamous epithelium with a fibrovascular connective tissue. Areas of pseudostratified ciliated columnar epithelium were also evident in some areas. Presence of sebaceous glands also seen. Based on these findings a confirmatory diagnosis of sublingual dermoid cyst with respiratory epithelium and submental dermoid cyst was made. Conclusion This report throws light on midline lesions of the oral cavity and hopes to add this rare case into the current differential diagnosis of the same.
Collapse
Affiliation(s)
- Manikandhan Ramanathan
- 1Department of Oral and Maxillofacial Surgery, Meenakshi Ammal Dental College and Hospital, Chennai, Tamil Nadu India
| | - Sivapathasundharam Balasundharam
- 2Department of Oral Pathology and Microbiology, Meenakshi Ammal Dental College and Hospital, Alapakkam Main Road, Maduravoyal, Chennai, Tamil Nadu 600095 India
| | - Amelia Christabel
- 3Cleft and Craniofacial Unit, Meenakshi Ammal Dental College and Hospital, Chennai, Tamil Nadu India
| | - Preethi Murali
- 2Department of Oral Pathology and Microbiology, Meenakshi Ammal Dental College and Hospital, Alapakkam Main Road, Maduravoyal, Chennai, Tamil Nadu 600095 India
| | - Sneha Pandem
- 3Cleft and Craniofacial Unit, Meenakshi Ammal Dental College and Hospital, Chennai, Tamil Nadu India
| |
Collapse
|
|
16
|
Histopathologic Finding of Both Gastric and Respiratory Epithelia in a Lingual Foregut Cyst. Case Rep Med 2015; 2015:278376. [PMID: 26294913 PMCID: PMC4534602 DOI: 10.1155/2015/278376] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/25/2015] [Accepted: 07/16/2015] [Indexed: 11/17/2022] Open
Abstract
Foregut cysts are uncommon, mucosa-lined congenital lesions that may occur anywhere along the gastrointestinal or respiratory tract and typically present within the first year of life. Although infrequent, these cysts may generate feeding or respiratory difficulties depending on the size and location of the lesion. Foregut cysts of the oral cavity are rarely seen and of those cases localized to the tongue are even more uncommon. We describe a 4-month-old girl with a foregut cyst involving the floor of mouth and anterior tongue. Subsequent histologic analysis demonstrated a cyst lined with both gastric and respiratory epithelia. This case represents an extremely rare finding of both gastric and respiratory epithelia lined within a single cystic structure in the tongue. Although a very rare finding, a foregut cyst should be on the differential diagnosis of any lesion involving the floor of mouth or tongue in an infant or child.
Collapse
|
|
17
|
Nayak DR, Bhandarkar AM, Joy J, Pai K. Neonatal lingual choristoma with thyroid hemiagenesis. BMJ Case Rep 2015; 2015:bcr-2015-209744. [PMID: 25948857 DOI: 10.1136/bcr-2015-209744] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/12/2022] Open
Abstract
A 45-day-old infant presented with choking spells and cyanosis. Examination revealed a lingual cyst. Contrast-enhanced CT confirmed the diagnosis of lingual cyst with incidental thyroid hemiagenesis. The child underwent excision of the lesion, which was reported as lingual choristoma.
Collapse
Affiliation(s)
- Dipak Ranjan Nayak
- Department of Otolaryngology-Head and Neck Surgery, Kasturba Medical College, Manipal University, Manipal, Karnataka, India
| | - Ajay M Bhandarkar
- Department of Otolaryngology-Head and Neck Surgery, Kasturba Medical College, Manipal University, Manipal, Karnataka, India
| | - Jasmi Joy
- Department of Otolaryngology-Head and Neck Surgery, Kasturba Medical College, Manipal University, Manipal, Karnataka, India
| | - Kanthilatha Pai
- Department of Pathology, Kasturba Medical College, Manipal University, Manipal, Karnataka, India
| |
Collapse
|
|
18
|
Enteric duplication cyst located at the posterior tongue: a rare case report and review of the literature. Case Rep Otolaryngol 2015; 2015:951878. [PMID: 25802786 PMCID: PMC4352945 DOI: 10.1155/2015/951878] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/04/2015] [Revised: 02/03/2015] [Accepted: 02/12/2015] [Indexed: 11/24/2022] Open
Abstract
The lingual localization of an enteric duplication is extremely rare but may present with respiratory and feeding problems that require emergency intervention. A 7-month-old boy was brought to our clinic with feeding difficulties and tongue swelling. Physical examination showed a cystic lesion located near the left side of the tongue base that caused tongue protrusion to the contralateral side. During surgery, a 3-cm diameter opaque thick-walled cyst was found to be very closely adherent to the base of tongue, which was excised in its entirety. Following surgery, the patient fed during the early postoperative period and no complications were observed other than hypersalivation. On histological examination, a cystic lesion lined with intestinal mucosa and goblet cells was found. We present the rare case of a duplication cyst of the posterior tongue, with a literature review.
Collapse
|
|
19
|
Kwak EJ, Jung YS, Park HS, Jung HD. Oral foregut cyst in the ventral tongue: a case report. J Korean Assoc Oral Maxillofac Surg 2014; 40:313-5. [PMID: 25551098 PMCID: PMC4279972 DOI: 10.5125/jkaoms.2014.40.6.313] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/13/2014] [Revised: 09/06/2014] [Accepted: 09/18/2014] [Indexed: 11/20/2022] Open
Abstract
An oral foregut cyst is a rare congenital choristoma lined by the respiratory and/or gastrointestinal epithelium. The exact etiology has not been fully identified, but it is thought to arise from misplaced primitive foregut. This lesion develops asymptomatically but sometimes causes difficulty in swallowing and pronunciation depending on its size. Thus, the first choice of treatment is surgical excision. Surgeons associated with head and neck pathology should include the oral foregut cyst in the differential diagnosis for ranula, dermoid cyst, thyroglossal duct cyst and lymphangioma in cases of pediatric head and neck lesions.
Collapse
Affiliation(s)
- Eun-Jung Kwak
- Department of Oral and Maxillofacial Surgery, Yonsei University College of Dentistry, Seoul, Korea
| | - Young-Soo Jung
- Department of Oral and Maxillofacial Surgery, Yonsei University College of Dentistry, Seoul, Korea. ; Oral Science Research Institute, Yonsei University College of Dentistry, Seoul, Korea
| | - Hyung-Sik Park
- Department of Oral and Maxillofacial Surgery, Yonsei University College of Dentistry, Seoul, Korea. ; Oral Science Research Institute, Yonsei University College of Dentistry, Seoul, Korea
| | - Hwi-Dong Jung
- Department of Oral and Maxillofacial Surgery, Yonsei University College of Dentistry, Seoul, Korea. ; Oral Science Research Institute, Yonsei University College of Dentistry, Seoul, Korea
| |
Collapse
|
|
20
|
Kün-Darbois JD, Breheret R, Bizon A, Paré A, Laccourreye L. Bronchogenic cyst of the tip of the tongue: report of two cases. Eur Ann Otorhinolaryngol Head Neck Dis 2014; 132:49-51. [PMID: 24993780 DOI: 10.1016/j.anorl.2013.11.007] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/07/2013] [Revised: 09/21/2013] [Accepted: 11/26/2013] [Indexed: 11/26/2022]
Abstract
INTRODUCTION Lingual bronchogenic cyst is a rare congenital malformation, classified as a choristoma. It is lined by respiratory epithelium. Extrathoracic sites are rare and the tongue is an exceptional site. CASE REPORT The authors report two cases of bronchogenic cyst of the tip of the tongue in newborn infants. These two initially asymptomatic lesions gradually increased in size, leading to feeding difficulties in one case. MRI demonstrated well-demarcated, solitary, unilocular cystic images. Surgical resection was easily performed and histological examination concluded on bronchogenic cyst. An aberrant right upper lobe tracheal bronchus was also present in one case. DISCUSSION Lingual choristomas are rare and defined histologically according to the type of epithelium. The embryological origin of lingual bronchogenic cysts remains controversial. MRI is the imaging examination of choice to characterize these lesions, but the definitive diagnosis is based on histological examination. Early, systematic surgical enucleation of all congenital lingual cysts must be performed to prevent obstructive and infectious complications.
Collapse
Affiliation(s)
- J-D Kün-Darbois
- Service de chirurgie maxillo-faciale et stomatologie, CHU d'Angers, 4, rue Larrey, 49100 Angers, France.
| | - R Breheret
- Service d'ORL et chirurgie cervico-faciale, CHU d'Angers, 4, rue Larrey, 49100 Angers, France
| | - A Bizon
- Service d'ORL et chirurgie cervico-faciale, CHU d'Angers, 4, rue Larrey, 49100 Angers, France
| | - A Paré
- Service de chirurgie maxillo-faciale et plastique de la face, CHU Trousseau, route de Loche, 37044 Tours cedex, France
| | - L Laccourreye
- Service d'ORL et chirurgie cervico-faciale, CHU d'Angers, 4, rue Larrey, 49100 Angers, France
| |
Collapse
|
|
21
|
Rosa ACG, Hiramatsu DM, de Moraes FRR, Passador-Santos F, de Araújo VC, Soares AB. Oral foregut cyst in a neonate. J Craniofac Surg 2013; 24:2158-2160. [PMID: 24220427 DOI: 10.1097/scs.0b013e31829af985] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/09/2023] Open
Abstract
Oral foregut cysts are congenital choristomas that arise in the oral cavity during embryonic development from remnants of foregut-derived epithelium. This is an unusual report of a neonate with a large congenital sublingual cystic lesion, extending superficially from the left ventral tongue to the anterior floor of the mouth, impeding breast-feeding. The differential diagnosis included dermoid cyst, epidermoid cyst, mucous retention cyst, and oral lymphangioma. The treatment of choice was enucleation under general anesthesia. Histology showed a cystic lesion with a ciliated pseudostratified columnar epithelium with numerous goblet cells. Immunohistochemistry was positive for cytokeratin 7 and thyroid transcription factor 1 and negative for cytokeratin 20, resulting in a final diagnosis of an oral foregut cyst. Three weeks after surgery, the tongue had healed with good mobility, and breast-feeding could be established. No recurrence was present at 6 months of follow-up.
Collapse
Affiliation(s)
- Ana Cláudia Garcia Rosa
- From the *Federal University of Tocantins, Palmas, Tocantins, †São Leopoldo Mandic Institute and Research Center, Campinas, São Paulo; ‡General Public Hospital of Palmas; and §Dona Regina Public Maternity Hospital, Palmas, Tocantins, Brazil
| | | | | | | | | | | |
Collapse
|
|
22
|
Pentenero M, Marino R, Familiari U, Gandolfo S. Choristoma involving the floor of the mouth and the anterior tongue: a case of teratoid cyst with gastric and respiratory epithelia. J Oral Maxillofac Surg 2013; 71:1706-11. [PMID: 23871469 DOI: 10.1016/j.joms.2013.04.020] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/04/2013] [Revised: 04/18/2013] [Accepted: 04/18/2013] [Indexed: 12/28/2022]
Abstract
Oral dysontogenic cysts result from defective embryonic development. Among them teratoid cysts are the most unusual presentation and may be lined by gastric, intestinal, respiratory, squamous, ciliated epithelium or even pancreatic structures. Teratoid cysts containing respiratory and gastrointestinal epithelium have typically been called choristomas. This article describes a 15-year-old boy presenting a choristoma involving both the floor of the mouth and the anterior tongue and characterized by the presence of squamous epithelium with skin adnexa, gastric and respiratory epithelium.
Collapse
Affiliation(s)
- Monica Pentenero
- Academic Researcher, Department of Oncology, Unit of Oral Medicine and Oral Oncology, University of Turin, Turin, Italy.
| | | | | | | |
Collapse
|
|
23
|
Joshi R, Cobb ARM, Wilson P, Bailey BMW. Lingual cyst lined by respiratory and gastric epithelium in a neonate. Br J Oral Maxillofac Surg 2012; 51:173-5. [PMID: 22554694 DOI: 10.1016/j.bjoms.2012.04.003] [Citation(s) in RCA: 15] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/12/2012] [Accepted: 04/10/2012] [Indexed: 10/28/2022]
Abstract
Congenital oral cysts of foregut origin include bronchogenic, enteric, and oesophageal cysts, and they are much rarer than the well described dermoid, epidermoid, and thyroglossal cysts. The exact aetiology is poorly understood, but they are thought to arise from misplaced embryonic rests of the primitive foregut. The presentation of cysts lined by respiratory or gastrointestinal epithelium in the oral cavity is unusual. There have been previous reports of bronchogenic or gastrointestinal epithelium-lined lingual cysts, but few report both features occurring within the same cyst. In view of the scarcity of such reports, we present the case of a lesion on the ventral surface of the tongue of a newborn boy. On removal it was found to be a cyst lined by immature squamous, respiratory, and gastric body epithelium.
Collapse
Affiliation(s)
- Rajnish Joshi
- Maxillofacial Unit, St Georges Hospital NHS Trust, London, United Kingdom
| | | | | | | |
Collapse
|
|
24
|
Batra R. The pathogenesis of oral choristomas. JOURNAL OF ORAL AND MAXILLOFACIAL SURGERY MEDICINE AND PATHOLOGY 2012. [DOI: 10.1016/j.ajoms.2011.11.005] [Citation(s) in RCA: 15] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 10/14/2022]
|
|
25
|
[Tongue cyst in children: foregut duplication, a possible diagnosis]. ACTA ACUST UNITED AC 2012; 113:442-7. [PMID: 22342887 DOI: 10.1016/j.stomax.2012.01.001] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/20/2011] [Revised: 08/29/2011] [Accepted: 01/02/2012] [Indexed: 11/24/2022]
Abstract
INTRODUCTION Foregut duplication is a heterotopy of the digestive mucosa. The tongue localization is relatively uncommon, it presents as a cystic lesion. We report a series of five patients presenting with cysts of the tongue. PATIENTS AND METHODS This retrospective study was made on cases of foregut duplication of the lingual area, diagnosed between 1977 and 2008. We documented the patient's age, gender, symptoms, lesion localization, clinical radiological and pathological features, treatment, and outcome. RESULTS Four boys and one girl were included. Two cysts were diagnosed during antenatal screening, two during early infancy, and one at the age of 12 (after infectious complication). In every case, the diagnosis could be confirmed only after surgical removal. DISCUSSION Foregut duplication of the tongue is rare and its incidence is probably underestimated. This diagnosis should be suggested in case of congenital intraoral cysts. Treatment is surgical excision, and pathological examination confirms the diagnosis.
Collapse
|
|
26
|
Toribio Y, Roehrl MH. The Nasolabial Cyst: A Nonodontogenic Oral Cyst Related to Nasolacrimal Duct Epithelium. Arch Pathol Lab Med 2011; 135:1499-503. [DOI: 10.5858/arpa.2010-0338-rs] [Citation(s) in RCA: 20] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/06/2022]
Abstract
Nasolabial cysts are interesting, relatively uncommon benign extraosseous maxillary lesions. We review current knowledge about epidemiology, symptoms, imaging modalities, pathogenesis, histopathologic and ultrastructural features, treatment options, and prognosis. Nasolabial cyst lining epithelium is characteristically composed of a basal layer of cuboidal cells and a luminal layer of columnar secretory cells with interspersed mucous goblet cells. In addition, areas of multilayered epithelium and squamous metaplasia may be seen. The cyst stroma is characterized by collagen-rich fibrovascular tissue with variably admixed chronic inflammatory cells. Furthermore, to our knowledge, we report the first example of immunohistochemical protein expression profiling of nasolabial cyst lining epithelium, discovering that basal layer cells express p63 and cytokeratin 5/6, while goblet cells express MUC-2 and MUC-5AC mucins, supporting the notion that nasolabial cysts can be understood as hamartomatous, locally expansile remnants of distal nasolacrimal duct development.
Collapse
|
|
27
|
Chai RL, Ozolek JA, Branstetter BF, Mehta DK, Simons JP. Congenital choristomas of the oral cavity in children. Laryngoscope 2011; 121:2100-6. [PMID: 21826675 DOI: 10.1002/lary.21758] [Citation(s) in RCA: 19] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/24/2010] [Accepted: 01/21/2011] [Indexed: 11/12/2022]
Abstract
OBJECTIVES/HYPOTHESIS To review our institutional experience with oral cavity choristomas in children. STUDY DESIGN Retrospective case series and medical record review. METHODS Medical records including clinic notes, operative reports, radiologic studies, and pathology specimens were reviewed. All imaging studies and pathology material were reviewed by a head and neck radiologist and pediatric pathologist, respectively. RESULTS Sixteen patients (10 males and 6 females) with congenital oral cavity choristomas were identified. Mean age at diagnosis was 1.8 months. Location of the lesions included tongue (n = 9) and floor of mouth (n = 7). Preoperative imaging included magnetic resonance imaging (n = 6), computed tomography (n = 5), plain radiography (n = 1), and no imaging (n = 4). Radiographically, the lesions were consistently lobular with well-defined margins, but other imaging features often mimicked other masses that can arise in the tongue and floor of mouth. Symptoms were present in five of 16 patients and included difficulty feeding, swelling with upper respiratory infection, and partial airway obstruction. Complete surgical excision was performed in 15 of 16 patients; mean age at the time of surgery was 12.7 months. One patient underwent marsupialization. No complications were noted perioperatively. No recurrences of choristoma were seen. On histologic examination, the predominant component was cystic with cyst linings of respiratory epithelium (n = 5), gastric (foveolar) epithelium (n = 1), or both (n = 10). CONCLUSIONS This study supports surgical excision as an effective intervention for children with oral cavity choristomas. Because the etiology of these cysts is unknown and diagnostic terminology is widely variable, we propose a more descriptive diagnosis based on the histology (i.e., lingual choristoma [or lingual developmental cyst] with respiratory epithelium or lingual choristoma [or lingual developmental cyst] with gastric epithelium).
Collapse
Affiliation(s)
- Raymond L Chai
- Department of OtolaryngologyChildren's Hospital of Pittsburgh of UPMC, University of Pittsburgh School of Medicine, Pittsburgh, Pennyslvania 15224, USA
| | | | | | | | | |
Collapse
|
|
28
|
Houshmand G, Hosseinzadeh K, Ozolek J. Prenatal magnetic resonance imaging (MRI) findings of a foregut duplication cyst of the tongue: value of real-time MRI evaluation of the fetal swallowing mechanism. JOURNAL OF ULTRASOUND IN MEDICINE : OFFICIAL JOURNAL OF THE AMERICAN INSTITUTE OF ULTRASOUND IN MEDICINE 2011; 30:843-850. [PMID: 21633000 DOI: 10.7863/jum.2011.30.6.843] [Citation(s) in RCA: 14] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 05/30/2023]
Abstract
Foregut duplication cysts of the oral cavity or lingual choristomas have a potential risk of airway obstruction. Two cases are reported that were initially detected by screening sonography. Further imaging with both static and real-time cine magnetic resonance imaging confirmed the lingual origin, relationship of the mass to fluid-filled spaces within the oral cavity, motion of the mass with the tongue during fetal swallowing, and airway patency. The additional information provided by magnetic resonance imaging aided in planning delivery and obviated the need for an ex utero intrapartum treatment procedure because airway patency was confirmed in both cases.
Collapse
Affiliation(s)
- Golbahar Houshmand
- Department of Radiology, University of Pittsburgh Medical Center, Pittsburgh, PA, USA
| | | | | |
Collapse
|
|
29
|
Abstract
A rare case of gastric duplication cyst of tongue is reported.
Collapse
|
|
30
|
Patel P, Branstetter BF, Myers EN. Lingual foregut duplication in a middle-aged adult. AJNR Am J Neuroradiol 2011; 32:E40-1. [PMID: 20075097 DOI: 10.3174/ajnr.a1994] [Citation(s) in RCA: 10] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/26/2022]
Abstract
Lingual duplication cysts are rare in the pediatric population and exceedingly rare in adults. Imaging is an important part of the evaluation of lingual lesions and is helpful in defining the location and extent for surgical planning. We present a lingual duplication cyst and discuss the imaging findings and radiologic differential diagnosis.
Collapse
Affiliation(s)
- P Patel
- Department of Radiology and Otolaryngology, University of Pittsburgh Medical Center, Pennsylvania, USA
| | | | | |
Collapse
|
|
31
|
Juneja M, Boaz K, Srikant N, Nandita KP, Vidya M. Lingual cyst lined by respiratory epithelium: a case report and review of literature. Fetal Pediatr Pathol 2011; 30:225-32. [PMID: 21434827 DOI: 10.3109/15513815.2011.555807] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/13/2022]
Abstract
The present report describes a case of a lingual cyst lined by a respiratory epithelium occurring in a 1-year-old girl. The article also reviews the cases published in the literature under the heading of lingual cysts and segregates all the cases having predominant respiratory epithelium as the cystic lining. Reclassification, with more descriptive histologic terminology to name the lingual cysts, has been proposed.
Collapse
Affiliation(s)
- Manish Juneja
- Department of Oral Pathology & Microbiology, Manipal College of Dental Sciences, Mangalore, Karnataka, India.
| | | | | | | | | |
Collapse
|
|
32
|
[Bronchogenic cyst of the tongue in an adult]. ACTA ACUST UNITED AC 2010; 111:46-8. [PMID: 20070994 DOI: 10.1016/j.stomax.2009.10.005] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/14/2009] [Revised: 10/22/2009] [Accepted: 10/23/2009] [Indexed: 01/11/2023]
Abstract
INTRODUCTION Choristoma are non-malignant embryonic tumors, defined by ectopic tissues different from the organ on which they develop. The bronchogenic cyst is a choristoma affecting the upper aero digestive tract. It is lined with respiratory epithelium. The tongue is a rare localization for choristoma. CASE REPORT We report the case of a 22-year old male patient who consulted in maxillofacial surgery for painful macroglossia. Imaging revealed a median lesion, suggesting an abscessed embryonic tumor. Pathologic examination after surgical exeresis confirmed the diagnosis of lingual choristoma of the bronchogenic cyst type. DISCUSSION Embryonic tumors of the tongue are generally diagnosed during childhood. Adult cases are rare. Surgical removal is the only treatment. Post-operative complications and relapse are rare. Only one case of malignant transformation has been reported; systematic radical exeresis is thus always recommended.
Collapse
|
|
33
|
Boffano P, Zavattero E, Campisi P, Gallesio C. Surgical treatment of an oral cyst with respiratory epithelium. J Craniofac Surg 2009; 20:1275-1277. [PMID: 19625847 DOI: 10.1097/scs.0b013e3181ae1794] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/25/2022] Open
Abstract
Cysts with respiratory epithelium are rare entities in the oral cavity. In the literature, there are only few cases of oral cystic masses lined by respiratory epithelium but lacking gastrointestinal epithelium. In this paper, we present the surgical treatment of a cyst with respiratory epithelium in the floor of the mouth affecting a 35-year-old woman. The asymptomatic lesion was noticed 6 years previously. On histopathologic examination, the cyst showed walls that were composed of connective tissue covered by pseudostratified ciliated columnar epithelium, interspersed by a few goblet cells. Oral cysts lined by respiratory epithelium are benign lesions. Cure is effected by surgical excision. This lesion should be considered in the differential diagnosis of masses involving the anterior tongue or the floor of the mouth.
Collapse
Affiliation(s)
- Paolo Boffano
- Division of Maxillofacial Surgery, Head and Neck Department, San Giovanni Battista Hospital, Turin, Italy.
| | | | | | | |
Collapse
|
|
34
|
Gopalakrishnan R, Koutlas IG, Schauer GM, Schnitker G. Dental (odontogenic) choristoma of the tongue. J Oral Maxillofac Surg 2009; 67:1135-8. [PMID: 19375030 DOI: 10.1016/j.joms.2008.08.020] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/12/2008] [Revised: 06/27/2008] [Accepted: 08/20/2008] [Indexed: 01/03/2023]
Affiliation(s)
- Rajaram Gopalakrishnan
- Division of Oral and Maxillofacial Pathology, University of Minnesota, Minneapolis, MN 55455, USA
| | | | | | | |
Collapse
|
|
35
|
Azañero WD, Mazzonetto R, León JE, Vargas PA, Lopes MA, de Almeida OP. Lingual cyst with respiratory epithelium: a histopathological and immunohistochemical analysis of two cases. Int J Oral Maxillofac Surg 2009; 38:388-92. [PMID: 19217261 DOI: 10.1016/j.ijom.2009.01.003] [Citation(s) in RCA: 16] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/25/2008] [Revised: 06/22/2008] [Accepted: 01/19/2009] [Indexed: 10/21/2022]
Abstract
Cysts of the tongue are rare, usually derived from epithelia of the embryonic gastrointestinal and respiratory tracts, and classified according to the predominant epithelium lining. These cysts are usually discovered during infancy, more frequently in males, but they may not appear until well into adulthood. The authors report two lingual cysts lined mainly with respiratory, and focally by squamous, epithelium. Periodic acid-Schiff and mucicarmine staining revealed focal positivity in intracystic mucoid material and goblet cells. Immunohistochemical analysis with vimentin, cytokeratins (AE1/AE3, 34betaE12, CK1, CK5, CK6, CK7, CK8, CK10, CK13, CK14, CK16, CK18, and CK19), E-cadherin, beta-catenin, and epithelial membrane antigen showed a similar profile of normal respiratory epithelium, suggesting well-differentiated states. Owing to their controversial origin, these cysts should be named descriptively, as suggested by Manor et al., as lingual cysts with respiratory epithelium.
Collapse
Affiliation(s)
- W D Azañero
- Department of Oral Medicine, Oral Surgery and Oral Pathology, Faculty of Stomatology, University Cayetano Heredia, 430, Lima, Perú.
| | | | | | | | | | | |
Collapse
|
|
36
|
Horie N, Shimoyama T, Kawano R, Ide F. Lateral lingual cyst with respiratory epithelium: a rare case. ACTA ACUST UNITED AC 2008. [DOI: 10.1111/j.1752-248x.2008.00027.x] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/01/2022]
|
|
37
|
Mahajan V, Mathew JL, Singh M, Gupta R, Das A. Vallecular cyst-revisited. Indian J Pediatr 2008; 75:1081-2. [PMID: 18810338 DOI: 10.1007/s12098-008-0190-7] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/15/2007] [Accepted: 03/25/2008] [Indexed: 12/01/2022]
Abstract
We report a 3 months-old-male infant presented with recurrent cough, noisy breathing and regurgitation of feeds since 15 days of life. Examination revealed inspiratory stridor. CECT showed cystic lesion in base of tongue. After excision it was proved a case of vallecular cyst on histopathology. Although rare, vallecular cyst should be included in the differential diagnosis of congenital laryngeal stridor in neonates. The literature of vallecular cyst is being reviewed in the current article.
Collapse
Affiliation(s)
- Vidushi Mahajan
- Post Graduate Institute of Medical Education and Research, Chandigarh, India
| | | | | | | | | |
Collapse
|
|
38
|
Benhammou A, Benyoussef N, Bencheikhe R, Benbouzid MA, Boulaich M, Essakali L, Kzadri M. [Bronchogenic cyst of the tongue]. ACTA ACUST UNITED AC 2006; 123:148-51. [PMID: 16840904 DOI: 10.1016/s0003-438x(06)76656-7] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/21/2022]
Abstract
OBJECTIVES Bronchogemc cyst is a rare aberration secondary to abnormal detachment of accessory lung buds from the primitive foregut. Through one case of bronchogenic cyst of the tongue, the authors made a review of this exceptional localisation. MATERIAL AND METHODS We report one case of bronchogenic cyst of the tongue treated in the Ibn Sina Hospital. RESULTS Our patient was a new born admitted in our formation for a voluminous cystic mass ot the tongue, responsible of difficulty for mouth occlusion and feeding The radiology I confirms the liquid nature of this mass. The treatment was surgical, and the diagnosis of bronchogenic cyst was histological. CONCLUSION Bronchogenic cysts are generally intrathoracic. The localization in the cervico-facial area is rare, and only 4 cases of bronchogenic cyst of the tongue were described in the literature. The clinical presentation is non specific, depending on the volume of the cyst. Radiology permits to confirm the nature of the mass. The certitude diagnosis is histological: bronchogenic cysts are lined by pseudostratified ciliated columnar epithelium with accessory tissues in the cyst wall, smooth muscle, seromucinous glands and cartilage, simulating a bronchus. Complete surgical excision is the base of the treatment.
Collapse
Affiliation(s)
- A Benhammou
- Service d'ORL et de Chirurgie Cervico-Faciale, CHU Rabat Salé, Maroc.
| | | | | | | | | | | | | |
Collapse
|
|
39
|
Burkart CM, Brinkman JA, Willging JP, Elluru RG. Lingual cyst lined by squamous epithelium. Int J Pediatr Otorhinolaryngol 2005; 69:1649-53. [PMID: 15955574 DOI: 10.1016/j.ijporl.2005.03.051] [Citation(s) in RCA: 18] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/31/2005] [Accepted: 03/29/2005] [Indexed: 11/30/2022]
Abstract
Lingual choristomas or foregut duplication cysts are infrequently reported congenital anomalies that typically present in the perinatal period, and can cause respiratory or feeding problems. These cysts are lined by epithelial cells characteristic of the upper aerodigestive tract. We describe two children who presented shortly after birth with lingual cysts lined with squamous epithelium and foci of respiratory epithelium. Unlike previously reported lingual choristomas, these cysts did not contain gastrointestinal epithelium. In both these cases, the lingual cysts were successfully excised via a transoral approach. This article will discuss the histological findings in these two cases, the differential diagnosis and attempt to classify these lesions in the context of existing terminology.
Collapse
Affiliation(s)
- Collin M Burkart
- University of Cincinnati College of Medicine, Department of Otolaryngology-HNS, Cincinnati, OH, USA
| | | | | | | |
Collapse
|
|
40
|
Ech-Cherif El Kettani N, Dafiri R. [Bronchogenic cyst of the tongue: a very rare localization]. JOURNAL DE RADIOLOGIE 2004; 85:1736-8. [PMID: 15669570 DOI: 10.1016/s0221-0363(04)97741-4] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 05/01/2023]
Abstract
Bronchogenic cysts are extremely rare in the tongue. We explain this localization by abnormal detachment of accessory lung buds from the primitive foregut. MR is the best imaging modality to evaluate lesions of tongue. Enucleation is the best treatment option. We present and discuss the case of a six month old infant presenting with a congenital tongue mass. This mass was a bronchogenic cyst.
Collapse
|
|
41
|
Robson CD. Cysts and tumors of the oral cavity, oropharynx, and nasopharynx in children. Neuroimaging Clin N Am 2003; 13:427-42, ix. [PMID: 14631683 DOI: 10.1016/s1052-5149(03)00038-8] [Citation(s) in RCA: 12] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/30/2022]
Abstract
A wide variety of developmental and neoplastic lesions arise in the oral cavity, oropharynx, and nasopharynx in children. The clinical manifestations and cross-sectional imaging findings provide complementary information, which is used to establish a probable or definitive diagnosis. This article describes imaging techniques and findings for developmental cystic masses and solid tumors in the pediatric population.
Collapse
Affiliation(s)
- Caroline D Robson
- Division of Neuroradiology, Department of Radiology, Children's Hospital, Harvard Medical School, 300 Longwood Avenue, Boston, MA 02115, USA.
| |
Collapse
|
|
42
|
Desuter G, Plouin-Gaudon I, de Toeuf C, Gosseye S, Hamoir M. Gastric choristoma of the midline neck in a newborn: a case report and review of the literature. J Pediatr Surg 2003; 38:E1-3. [PMID: 12861589 DOI: 10.1016/s0022-3468(03)00210-0] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/17/2022]
Abstract
The current report describes a 3-week-old boy presenting with a gastric choristoma of the anterior compartment of the neck. Initial examination at birth found 3 masses in the anterior region of the neck. The only remarkable symptom consisted of episodes of cyanosis induced by neck flexion. The neck mass was removed after computed tomography scan assessment. Pathology finding showed the diagnosis of gastric choristoma. Cervical location of a gastric choristoma in children is rare. Its presentation as a clinically palpable anterior compartment cervical mass at birth even more rare.
Collapse
Affiliation(s)
- G Desuter
- Department of Otolaryngology-Head & Neck Surgery, St-Luc University Hospital, Université catholique de Louvain, Brussels, Belgium
| | | | | | | | | |
Collapse
|
|
43
|
Cuillier F, Samperiz S, Testud R, Fossati P. Antenatal diagnosis and management of a vallecular cyst. ULTRASOUND IN OBSTETRICS & GYNECOLOGY : THE OFFICIAL JOURNAL OF THE INTERNATIONAL SOCIETY OF ULTRASOUND IN OBSTETRICS AND GYNECOLOGY 2002; 20:623-626. [PMID: 12493055 DOI: 10.1046/j.1469-0705.2002.00860.x] [Citation(s) in RCA: 17] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 05/24/2023]
Abstract
Vallecular cysts are rare. It is important to be aware of their presence as they can cause upper airway obstruction. We describe a case of congenital vallecular cyst diagnosed at 28 weeks of gestation. This early diagnosis enabled us to plan careful perinatal management. We believe that, in cases of suspected oropharyngeal tumors, elective delivery should be carried out in a tertiary referral center in which emergency ventilation and tracheostomy are possible.
Collapse
|