Neonatal Permanent Jaw Constriction Because of Oral Synechiae and Pierre Robin Sequence in a Child with van der Woude Syndrome

Denion, É.; Capon, Norman B.; Martinot, V.; Pellerin, P

doi:10.1597/1545-1569_2002_039_0115_npjcbo_2.0.co_2

Cited by 12 publications

(11 citation statements)

References 10 publications

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“…The patient in our case did not have an extensive web behind the knee, which is the typical feature of popliteal pterygium syndrome. van der Woude syndrome is also associated with intraoral synechiae and a cleft palate (Valnicek and Clarke, 1993; Denion et al, 2002; Puvabanditsin et al, 2003; Robbins et al, 2019). Both LS (Denion et al, 2002) and AS (Valnicek and Clarke, 1993; Denion et al, 2002; Puvabanditsin et al, 2003) have been reported as intraoral synechiae associated with this syndrome; however, our patient did not have pits on the lower lip that are typically observed in this syndrome.…”

Section: Discussionmentioning

confidence: 99%

Congenital Lateral Palatal Synechia Associated With Cleft Palate: A Case Report With Long-Term Follow-Up and Review of the Literature

Imai

Tachi

2019

The Cleft Palate Craniofacial Journal

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Lateral palatal synechiae are rare congenital adhesions running from the free borders of the cleft palate to the lateral parts of the tongue or the oral cavity floor, typically found in cleft palate lateral synechiae syndrome. We present a case of congenital lateral palatal synechia associated with a cleft palate that we treated and followed up for 10 years. We present the long-term prognosis. We also discuss variations in intraoral synechiae associated with cleft palate and the etiology of lateral palatal synechiae through a literature review.

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Section: Discussionmentioning

confidence: 99%

Congenital Lateral Palatal Synechia Associated With Cleft Palate: A Case Report With Long-Term Follow-Up and Review of the Literature

Imai

Tachi

2019

The Cleft Palate Craniofacial Journal

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“…At present, there is no consistent gene locus linked to either cleft palate lateral synechia syndrome or congenital alveolar synechiae syndrome (Jaeger et al, 2003). The intraoral fibrous bands seen in VWS and PPS are due to genetic mutations affecting interferon regulatory factor 6 (Kondo et al, 2002; Denion et al, 2002). The 3 cases in our study did not have a family history of cleft.…”

Section: Discussionmentioning

confidence: 99%

Patients With Cleft Lip and Palate Associated With Intraoral Fibrous Bands: A Report of 3 Cases and Review of Literature

Vamvanij

Chen

2019

The Cleft Palate-Craniofacial Journal

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In rare instances, cleft lip and palate occur in association with synechia, intraoral fibrous bands connecting the maxilla and mandible. The main concern in synechia is a restricted mouth opening that leads to airway and feeding problems. This study reports our experience in the treatment of 3 cases and includes a review of the literature. Three patients with intraoral fibrous bands received treatment and follow-ups in our center. Division of the bands and reconstruction of the lip and palate were successfully performed. All reported cases in the literature were collected for a summary of clinical presentations and as references for management. Early management of the synechia improves the mouth opening and facilitates the treatment of associated anomalies.

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“…The patient described in this case has VWS along with oral synechiae and PRS, which rarely occur synchronously. Denion et al (2002) described the first reported case of a child with VWS, PRS, and oral synechiae. The patient in their report had fibrous intermaxillary synechiae and a subglossopalatal membrane.…”

Section: Discussionmentioning

confidence: 99%

Combined Tongue-Palate Fusion With Alveolar Bands in a Patient With Pierre Robin Sequence and Van der Woude Syndrome

Robbins

Zárate

Hartzell

2018

The Cleft Palate Craniofacial Journal

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This report describes the presentation of a newborn male with circumferential tongue-palate fusion associated with cleft palate and alveolar bands. After intraoral adhesions lysis, the patient was diagnosed with Pierre Robin sequence. A family history of cleft lip and palate was noted, and interferon regulatory factor 6 ( IRF6) sequencing revealed a heterozygous variant, confirming the diagnosis of van der Woude syndrome. The disruption of IRF6 resulted in abnormal orofacial development including micrognathia and intraoral adhesions as well as tongue-palate fusion, then resulting in glossoptosis with airway obstruction and cleft palate.

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Neonatal Permanent Jaw Constriction Because of Oral Synechiae and Pierre Robin Sequence in a Child with van der Woude Syndrome

Cited by 12 publications

References 10 publications

Congenital Lateral Palatal Synechia Associated With Cleft Palate: A Case Report With Long-Term Follow-Up and Review of the Literature

Congenital Lateral Palatal Synechia Associated With Cleft Palate: A Case Report With Long-Term Follow-Up and Review of the Literature

Patients With Cleft Lip and Palate Associated With Intraoral Fibrous Bands: A Report of 3 Cases and Review of Literature

Combined Tongue-Palate Fusion With Alveolar Bands in a Patient With Pierre Robin Sequence and Van der Woude Syndrome

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