Double Tongue,Intraoral Anomalies,and Cleft Palate—Case Reports and a Discussion of Developmental Pathology

Abstract

Objective

Isolated cleft palate is the most common presentation of the non-syndromic cleft lip/palate combinations and is multifactorial in etiology. We report two cases of children with clefts of the secondary palate coexistent with double tongue and in either case mandibular epulis or superiorly displaced salivary gland.

Results and Discussion

In each case, the palatal cleft correlated anatomically with the intraoral space–occupying lesion. The ratio of tongue volume to intraoral volume during palatogenesis is discussed with reference to the pathogenesis of cleft palate. These clinical cases propose the model of a unifying sequence of developmental events whereby deformation of palatal shelf elevation results in secondary palatal clefting.

Keywords

cleft palate developmental pathology double tongue epulis

References

Bartholdson

Hellstrom

S.O.

Soderberg

A case of double tongue. Case report.

Scand J Plast Reconstr Surg Hand Surg. 1991; 25: 93–95.

Bell

R.C.

A child with two tongues (oral facial digital syndrome).

Br J Plast Surg. 1971; 24: 193–196.

Britto

J.A.

Chan

J.C.

Evans

R.D.

Hayward

R.D.

Thorogood

Jones

B.M.

From genotype to phenotype—molecular studies in apert cleft palatogenesis. Presented at the British Association of Plastic Surgeons; December 1998; London, England.

Demuth

R.J.

Johns

D.F.

Recurrent aspiration pneumonitis in a cleft palate child with hamartoma of the tongue.

Cleft Palate J. 1981; 18: 299–303.

Diewert

V.M.

Differential changes in cartilage cell proliferation and cell density in the rat craniofacial complex during secondary palate development.

Anat Rec. 1980; 198: 219–228.

Diewert

V.M.

A comparative study of craniofacial growth during secondary palate development in four strains of mice.

J Craniofac Genet Dev Biol. 1982; 2: 247–263.

Diewert

V.M.

Craniofacial growth during human secondary palate formation, and potential relevance of experimental cleft palate observations.

J Craniofac Genet Dev Biol Suppl. 1986; 2: 267–276.

Diewert

V.M.

Tait

Palatal process movement in the rat as demonstrated in frozen sections.

J Anat. 1979; 128: 609–618.

Emmanouil-Nikoloussi

E.N.

Kerameos-Foroglou

Developmental malformations of human tongue and associated syndromes (review).

Bull Group Int Rech Sci Stomatol Odontol. 1992; 35: 5–12.

10.

Ferguson

M.W.

Developmental mechanisms in normal and abnormal palate formation with particular reference to the etiology, pathogenesis, and prevention of cleft palate.

Br J Orthod. 1981; 8: 115–137.

11.

Ferguson

M.W.

Palate development.

Development. 1998; 103 Suppl: 41–60.

12.

Ferguson

M.W.

Sharpe

P.M.

Thomas

B.L.

Beck

Differential expression of insulin-like growth factors I and II (IGF I and II) mRNA, peptide and binding protein 1 during mouse palate development: comparison with TGF beta peptide distribution.

J Anat. 1992; 181: 219–238.

13.

Hanna

Argenyi

Z.B.

Benda

J.A.

Hamartoma of the tongue in an infant with a primary diagnosis of ectrodactyly-ectodermal dysplasia-cleft lip and palate syndrome.

J Cutan Pathol. 1994; 21: 173–178.

14.

Ishii

Takemori

Suzuki

J.I.

Hamartoma of the tongue.

Arch Otolaryngol. 1968; 88: 79–81.

15.

Johnston

Nash

D.J.

Sagittal growth trends of the development of cleft palate in mice homozygous for the ‘paddle’ gene.

J Craniofac Genet Dev Biol. 1982; 2: 265–276.

16.

Kimes

K.R.

Mooney

M.P.

Seigel

M.I.

Todhunter

J.S.

Size and growth rate of the tongue in normal and cleft lip and palate human fetal specimens.

Cleft Palate Craniofac J. 1991; 28: 212–221.

17.

Koch

B.L.

Myer

3rd Egelhoff

J.C.

Congenital epulis.

Am J Neuroradiol. 1997; 18: 739–741.

18.

Larsen

W.J.

Human Embryology. New York: Churchill Livingstone; 1993.

19.

Lauder

J.M.

Zimmerman

E.F.

Sites of serotonin uptake in epithelia of the developing mouse palate, oral cavity, and face: possible role in morphogenesis.

J Craniofac Genet Dev Biol. 1988; 8: 265–276.

20.

Martinot

V.L.

Manouvrier

Anastassov

Ribiere

Pellerin

P.N.

Orodigitofacial syndromes type 1 and II: clinical and surgical studies.

Cleft Palate Craniofac J. 1994; 31: 401–408.

21.

Minami

Sugahara

Mori

Mishima

Ankyloglossia superior: report of a case.

J Oral Maxillofac Surg. 1995; 53: 588–589.

22.

Miyamoto

Nagayama

Hayashi

A cleft palate child with lobulated tongue and lingual hamartoma.

J Oral Maxillofac Surg. 1991; 49: 644–646.

23.

Mooney

M.P.

Seigel

M.I.

Kimes

K.R.

Todhunter

J.S.

Janosky

Multivariate analysis of second trimester midfacial morphology in normal and cleft lip and palate fetal specimens.

Am J Phys Anthropol. 1992; 88: 203–209.

24.

Perri

F.A.

Myoepithelial hamartoma of the tongue.

Arch Otolaryngol. 1956; 64: 289–290.

25.

Qiu

C.X.

Ferguson

M.W.

The distribution of PDGFs and PDGF receptors during murine secondary palate development.

J Anat. 1995; 186: 17–29.

26.

Sharpe

P.M.

Ferguson

M.W.

Mesenchymal influences on epithelial differentiation in developing systems.

J Cell Sci Suppl. 1988; 10: 195–230.

27.

Stamm

Tauber

Hamartoma of the tongue.

Laryngoscope. 1945; 55: 104–146.

28.

Stanier

Forbes

S.A.

Arnason

Bjornsson

Sveinbjornsdottir

Williamson

Moore

The localization of a gene causing X-linked cleft palate and ankyloglossia (CPX) in an Icelandic kindred is between DXS326 and DXYS1X.

Genomics. 1993; 17: 549–555.

29.

Ugras

Demirtas

Bekerecioglu

Kutluhan

Karakok

Peker

Immunohistochemical study on histogenesis of congenital epulis and review of the literature.

Pathol Int. 1997; 47: 627–632.

30.

Widgerow

A.D.

Klippel-Feil anomaly, cleft palate, and bifid tongue.

Ann Plast Surg. 1990; 25: 216–222.

31.

Young

Feeley

Shah

R.M.

Does the tongue play a role in the initial development of vertical palatal shelf in hamster?

J Craniofac Genet Dev Biol. 1991; 11: 146–155.

32.

Zimmerman

E.F.

Wee

E.L.

Phillips

Roberts

Presence of serotonin in the palate just prior to shelf elevation.

J Embryol Exp Morphol. 1981; 64: 233–250.