Embryologic Features of Term Fetuses and Newborns in CL/Fr Mice with Special Reference to Cyanosis

Abstract

Objective

The objective of this study was to clarify the embryologic features of CL/Fr mice in relation to cyanosis.

Method

Orofacial examinations were conducted on 18-day-old term fetuses and newborns. Except for open eyelids in a few cases, all of the external abnormalities in this strain were cleft lip and/or palate (CLP).

Results

The CLP incidence in term fetuses was 28.4%. The body and placental weights were not different between CLP(+) and CLP(-) fetuses. Several types of CLP were found; the most frequent was bilateral complete CLP (60%) followed by left complete (16%). Bilateral complete CL, which is the most severe CL type, was associated with poorly developed secondary palate and wide cleft. In newborns, 39.4% of CLP(+) mice showed cyanosis, where no CLP(-) mice had such signs. The body weight of cyanotic mice with CLP was significantly lighter than that of noncyanotic mice with CLP. Cyanotic mice had severe types of CLP and unelevated palatal shelves.

Conclusion

We suggest that mechanical airway obstruction may be one of the causes of cyanosis in newborn CL/Fr mice.

Keywords

airway obstruction CL/Fr mice cyanosis development of palatal shelves Pierre Robin sequence

Get full access to this article

View all access options for this article.

References

Asada

Experimental study on cleft lip of CL/Fr mouse using whole embryo culture system (in Japanese).

Kokubyo Gakkai Zasshi 1987; 54: 47–56.

Bornstein

, Trasler

D.G.

, Fraser

F.C.

Effect of the uterine environment on the frequency of spontaneous cleft lip in CL/Fr mice.

Teratology 1970; 3: 295–298.

Braunwald

Hypoxia, polycythemia and cyanosis. In: Isselbacher

K.J.

, Martin

J.B.

, Braunwald

eds. Harrison's principles of internal medicine. 13th Ed. New York: McGraw-Hill, 1994: 178–183.

Bronsky

P.T.

, Johnston

M.C.

, Sulik

K.K.

Morphogenesis of hypoxia-induced cleft lip in CL/Fr mice.

J Craniofac Genet Dev Biol 1986; 2: 113–128.

Brown

K.S.

, Hetzel

S.C.

, Harne

L.C.

, Long

Blebs and hematomas in the lips of CL/Fr and A/J mice.

J Craniofac Genet Dev Biol 1985; 1: 313–322.

Canick

L.M.

Cleft lip and cleft palate. A review of embryology, pathologic anatomy and etiology.

Plast Reconstr Surg 1954; 14: 30.

Davidson

J.G.

, Fraser

F.C.

, Schlager

A maternal effect on the frequency of spontaneous cleft lip in the A/J mouse.

Teratology 1969; 2: 371–376.

Esaki

Decrease in cleft lip incidence by urethane administration to CL/Fr mice.

Cong Anom 1983; 23: 395–398.

Fraser

F.C.

, Rosen

Association of cleft lip and atrial septal defect in mice: a preliminary report.

Teratology 1975; 11: 321–324.

10.

Horiuchi

, Natsume

, Miura

. Experimental study on the development of cleft lip and/or palate. Report 9: comparison of cleft patterns between human and mouse (in Japanese). J Jpn Cleft Palate Assoc 1990; 15: 164–170.

11.

Horiuchi

The morphological study on the development of cleft lips in spontaneous cleft lip and/or palate mice (CL/Fr and A/J strains) (in Japanese).

J Jpn Cleft Palate Assoc 1992; 17: 304–332.

12.

Imai

Growth and development of craniofacial complex of mice with spontaneous cleft lip and palate—from secondary palate formation to immediately before birth (in Japanese).

Kokubyo Gakkai Zasshi 1980; 47: 213–234.

13.

Juriloff

D.M.

, Fraser

F.C.

Genetic maternal effects on cleft lip frequency in A/J and CL/Fr mice.

Teratology 1980; 21: 167–175.

14.

Juriloff

D.M.

, Mah

D.G.

The major locus for multifactorial nonsyndromic cleft lip maps to mouse Chromosome 11.

Mamm Genome 1995; 6: 63–69.

15.

Kadowaki

, Kamiishi

Relationship between the cyanosis and congenital heart anomalies in newborns of CL/Fr mice (Abstract, in Japanese).

J Jpn Cleft Palate Assoc 1993; 18: 430.

16.

Kadowaki

, Sakamoto

, Tanimura

, Kamiishi

Embryological features with special reference of craniofacial development of term fetuses and newborns in CL/Fr mice (Abstract).

Teratology 1992; 46: 9B.

17.

Machida

The malformations combined with cleft lip and palate. In: Miyazaki

, ed. Cleft palate—basic and clinic (in Japanese). Tokyo: Ishiyaku Press, 1982: 69–79.

18.

Millicovsky

, Ambrose

L.J.H.

, Johnston

M.C.

Developmental alterations associated with spontaneous cleft lip and palate in CL/Fr mice.

Am J Anal 1982; 164: 29–44.

19.

Nakane

, Kameyama

Environmental teratological study on the manifestation of multifactorial malformations. 6. Effect of maternal administration of vitamin A on the manifestation of cleft lip in CL/Fr mice (in Japanese).

Ann Res Envir Med, Nagoya Univ 1987; 38: 229–230.

20.

Nakane

, Kameyama

Environmental teratological study on the manifestation of multifactorial malformations. 8.

Effect of maternal administration of vitamin A on the manifestation of cleft lip in CL/Fr mice (2nd report) (in Japanese), Ann Res Envir Med, Nagoya Univ 1989; 40: 250–251.

21.

Nakane

, Sano

, Kameyama

Frequency of spontaneous cleft lip in CL/Fr strain of mice (in Japanese).

Ann Res Envir Med, Nagoya Univ 1980; 31: 275–276.

22.

Narukawa

, Natsume

, Miura

, Kawai

Decrease in cleft lip and palate by vitamin B₁₂ in CL/Fr mice.

Aichi-Gakuin Dent Sci 1988; 1: 29–34.

23.

Natsume

, Narukawa

, Kawai

, Takahashi

Experimental study on cleft lip and palate II. Incidence of spontaneous cleft lip and palate in mice (CL/Fr strain) that we are maintaining (in Japanese).

J Jpn Cleft Palate Assoc 1986; 11: 172–177.

24.

Okada (Sano)

Cleft lip (CL/Fr mouse). In: Kawamata

, Matsushita

eds. Handbook of animal models of disease 2 (in Japanese). Tokyo: Ishiyaku Press, 1982: 412–416.

25.

Trasler

D.G.

, Fraser

F.C.

Role of the tongue in producing cleft palate in mice with spontaneous cleft lip.

Dev Biol 1963; 6: 45–60.

26.

Trasler

D.G.

, Leong

Mitotic index in mouse embryos with 6-aminoni-cotinamide-induced and inherited cleft lip.

Teratology 1982; 25: 259–265.

27.

Trasler

D.G.

, Ohannessian

Ultrastructure of initial nasal process cell fusion in spontaneous and 6-aminonicotinamide-induced mouse embryo cleft lip.

Teratology 1983; 28: 91–101.

28.

Tsuda

, Moriguchi

, Hamanaka

. (in Japanese). Statistical study of bilateral cleft lip and/or palate. J Jpn Cleft Palate Assoc 1995; 20: 215–219.

29.

van der Meulen

J.C.

, Mazzola

, Stricker

, Raphael

Classification of craniofacial malformations. In: Stricker

, van der Meulen

J.C.

, Raphael

eds. Craniofacial malformations. Edinburgh: Churchill Livingston, 1990: 278–279.

30.

Wang

K.Y.

, Diewert

V.M.

A morphometric analysis of craniofacial growth in cleft lip and noncleft mice, J Craniofac Genet Dev Biol 1992; 12: 141–154.