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Dermoid cyst of the floor of the mouth in an infant
J. max.-fac. Surg. 5 (1977) 211-213 © Georg Thieme Verlag, Stuttgart
Dermoid Cyst of the Floor of the Mouth in an Infant (Case Report) Ruth YESCHUA, Itai Arye BAB, Menachem Ron WEXLER, Zvi NEVMAN
Department of Plastic and Maxillofacial Surgery The Hebrew University-Haddassah Medical School and Department of Oral Pathology, The Hebrew University-Haddassah School of Dental Medicine, Jerusalem, Israel
Summary
Pathology
Dermoid cysts of the floor of the mouth in the infant are rarely encountered although the aetiology is mainly embryological. Meyer proposes the classification of the cysts based on the histological differentiation of the elements of the cyst wall. A case is reported which shows differences in the epithelial lining of the cysts found followed by the differential diagnosis and treatment.
The specimen obtained for histopathological examination consisted of three closely linked cystic structures, the largest measuring 3 cm in diameter, and the other two, 1 and 2 cm respectively. One of them was found to contain white milky fluid, while the other two were empty.
Key-Words:Dermoid cyst; Floor of the mouth; Infant. Introduction Dermoid cysts of the floor of the mouth in the infant are rarely encountered. Sporadic casereports have been published mostly in the Oral Surgery periodicals (Hooley et al. 1967~ Gold et aI. 1974, Hoggins 1974). Nevertheless the plastic surgeon may be confronted with such findings, which present interesting diagnostic and differential diagnostic problems. The treatment is always surgical.
Case Report A seven month old female infant was referred to our clinic with a sublingual swelling pushing the tongue upwards and causing difficulties in feeding. Tile parents were aware of this swelling since birth but only lately had it become more apparent. On physical examination the only positive finding was a mass of soft consistency, 4 × 4 x 4 cm, in size filling the floor of the mouth, pushing the tongue upwards towards the palate, and it could be transilluminated (Fig. 1). Under general anaesthesia and nasal intubation the lesion was enucleated via a sublinguaI transverse incision. Three consecutive cystic structures were removed (Fig. 2). The floor of the mouth was sutured and post operative recovery was uneventful.
The microscopic examination revealed two types of epithelium, lining the cystic lesions: Orthokeratinizing stratified squamous epithelium (Fig. 3) and nonkeratinizing stratified squamous epithelium (Fig. 4) showing marked hydropic degeneration in the stratum spinosum. The cysts were surrounded by a relatively thick layer of granulation tissue, scantily infiltrated by lymphocytes. The presence of hair follicles, sebaceous glands and occasionally sweat glands as well as adipose tissue was noted around the cysts, particularly those lined by a keratinizing epithelium. The lumen of the cysts lined by a keratinizing epithelium were filled with desquamated keratin squames (Fig. 3). The excretory ducts of the submandibular and sublingual salivary glands were also included in the specimen. They were lined either by a thin layer of stratified squamous epithelium or pseudostratifled ciliated columnar epithelium with Goblet cells. The diagnosis is consistent with that of a dermold cyst.
Discussion The occurrence of sublingual dermoid cysts is extremely rare in the infant and presents an interesting diagnostic problem. Only 6.9o/0 of the dermoid cysts found in humans were in the head and neck region (New and Erich 1987).
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R. Yeschua, I. A. Bab, M. R. WexIer, Z. Neuman
Fig. 1 A mass of soft consistency filling the floor of the mouth, pushing the tongue upwards.
Fig. 2 Following a transverse incision, three cystic formations appeared.
Fig. 3 Wall of dermoid cyst composed of keratinizing epithelium and granulation tissue. Sebaceous glands, hair follicles and a sweat gland are present in the granulation tissue. The part of the cyst lumen included in the photomicrograph is filled with desquamated keratin squames. HE, x28).
Fig. 4 Wall of cyst composed of nonkeratinizing stratified squamous epithelium with hydropic degeneration in the stratum spinosum and granulation tissue with a mild lymphocytic infiltration. HE, x64).
Dermoid Cyst of the Floor of the Mouth in an Infant Meyer (1955) suggested a classification of the
0ral cysts based on the histological findings: Epidermal cyst - The cyst wall is derived from epidermis and connective tissue. Dermoid cyst - Epithelial lined cavity with the presence of normal dermal appendages such as hair follicles and sebaceous or sweat glands in the connective tissue. Teratoma - Dermal appendages associated with mesodermal and endodermal derivatives such as osseous and dental structures and respiratory and intestinal epithelium. The term "Dermoid" was suggested as a clinical term for all developmental cysts of the floor of the mouth (Meyer 1955). A possible cause o f those formations is entrapment of epithelial debris or rests during the midline closure of the first and second branchial arches. Some of the cysts may be formed from remnants of the "tuberculum impar of His" which during the third and fourth week of intrauterine development will form, together with the inner surface of each mandibular arch, the body of the tongue and the floor of the mouth. Another possibility is the submergence of ecto-
213
dermal cells at the time of closure of the anterior neuropore. In the literature from 1949-1967 reviewed by Y o s h i m u r a et al. (1970) only 7 congenital cysts were included among reported cases. Clinically it is important to establish the relation of the cyst to the mylo-hyoid and genio-hyoid muscles which make the swelling appear in a sublingual or submental position. This determines whether the approach to the tumour should be intraorally or through an external incision. Sometimes a combined approach is necessary. The differential diagnosis is: Ranula (originating from Wharton's duct); thyroglossal duct cyst; cystic hygroma; branchial cleft cyst; acute infection or cellulitis of the submaxillary and sublingual glands or floor of the mouth; tumours of salivary glands; normal fat and lipomata. Conclusion
Dermoid cysts of the floor of the mouth are rare. The most interesting feature of the case presented is the difference in epithelial lining between the closely linked cystic structures. A difference in development can therefore be suspected.
References Gold, B. D., D. E. Sheinkop[, B. Levy: Dermoid, epi-
dermoid and teratomatous cysts of the floor of the mouth. J. oral Surg. 32 (1974) 2 Hoggins, G. S.: Congenital sublingual cystic swelling due to imperforate salivary duct. Oral Surg. 37 (1974) 370 Hooley, ]. R., G. L. Freedman, R, S. Redma,nn: Sublingual dermoid cyst in an infant. Oral Surg. 24 (1967) 715 Meyer, ],: Dermoid cysts of the floor of the mouth. Oral Surg. 8 (1955) 1149
New, G. B., J. B. Erich: Dermoid cysts of the head
and neck. Surg. Gynec. Obstet. 65 (1937) 48 Yoshimura, Y., K. Takada, M. Takeda, T. Mimura, M. Mori: Congenital dermoid cysts of the sub-
lingual region. Report of case. J. oral Surg. 28 (1970) 366 M. R. I4Zexler, M.D., Plastic Surgery, H.U.H. P.O. Box 499, ]erusalem/Israel
Dermoid Cyst of the Floor of the Mouth in an Infant (Case Report) Ruth YESCHUA, Itai Arye BAB, Menachem Ron WEXLER, Zvi NEVMAN
Department of Plastic and Maxillofacial Surgery The Hebrew University-Haddassah Medical School and Department of Oral Pathology, The Hebrew University-Haddassah School of Dental Medicine, Jerusalem, Israel
Summary
Pathology
Dermoid cysts of the floor of the mouth in the infant are rarely encountered although the aetiology is mainly embryological. Meyer proposes the classification of the cysts based on the histological differentiation of the elements of the cyst wall. A case is reported which shows differences in the epithelial lining of the cysts found followed by the differential diagnosis and treatment.
The specimen obtained for histopathological examination consisted of three closely linked cystic structures, the largest measuring 3 cm in diameter, and the other two, 1 and 2 cm respectively. One of them was found to contain white milky fluid, while the other two were empty.
Key-Words:Dermoid cyst; Floor of the mouth; Infant. Introduction Dermoid cysts of the floor of the mouth in the infant are rarely encountered. Sporadic casereports have been published mostly in the Oral Surgery periodicals (Hooley et al. 1967~ Gold et aI. 1974, Hoggins 1974). Nevertheless the plastic surgeon may be confronted with such findings, which present interesting diagnostic and differential diagnostic problems. The treatment is always surgical.
Case Report A seven month old female infant was referred to our clinic with a sublingual swelling pushing the tongue upwards and causing difficulties in feeding. Tile parents were aware of this swelling since birth but only lately had it become more apparent. On physical examination the only positive finding was a mass of soft consistency, 4 × 4 x 4 cm, in size filling the floor of the mouth, pushing the tongue upwards towards the palate, and it could be transilluminated (Fig. 1). Under general anaesthesia and nasal intubation the lesion was enucleated via a sublinguaI transverse incision. Three consecutive cystic structures were removed (Fig. 2). The floor of the mouth was sutured and post operative recovery was uneventful.
The microscopic examination revealed two types of epithelium, lining the cystic lesions: Orthokeratinizing stratified squamous epithelium (Fig. 3) and nonkeratinizing stratified squamous epithelium (Fig. 4) showing marked hydropic degeneration in the stratum spinosum. The cysts were surrounded by a relatively thick layer of granulation tissue, scantily infiltrated by lymphocytes. The presence of hair follicles, sebaceous glands and occasionally sweat glands as well as adipose tissue was noted around the cysts, particularly those lined by a keratinizing epithelium. The lumen of the cysts lined by a keratinizing epithelium were filled with desquamated keratin squames (Fig. 3). The excretory ducts of the submandibular and sublingual salivary glands were also included in the specimen. They were lined either by a thin layer of stratified squamous epithelium or pseudostratifled ciliated columnar epithelium with Goblet cells. The diagnosis is consistent with that of a dermold cyst.
Discussion The occurrence of sublingual dermoid cysts is extremely rare in the infant and presents an interesting diagnostic problem. Only 6.9o/0 of the dermoid cysts found in humans were in the head and neck region (New and Erich 1987).
212
R. Yeschua, I. A. Bab, M. R. WexIer, Z. Neuman
Fig. 1 A mass of soft consistency filling the floor of the mouth, pushing the tongue upwards.
Fig. 2 Following a transverse incision, three cystic formations appeared.
Fig. 3 Wall of dermoid cyst composed of keratinizing epithelium and granulation tissue. Sebaceous glands, hair follicles and a sweat gland are present in the granulation tissue. The part of the cyst lumen included in the photomicrograph is filled with desquamated keratin squames. HE, x28).
Fig. 4 Wall of cyst composed of nonkeratinizing stratified squamous epithelium with hydropic degeneration in the stratum spinosum and granulation tissue with a mild lymphocytic infiltration. HE, x64).
Dermoid Cyst of the Floor of the Mouth in an Infant Meyer (1955) suggested a classification of the
0ral cysts based on the histological findings: Epidermal cyst - The cyst wall is derived from epidermis and connective tissue. Dermoid cyst - Epithelial lined cavity with the presence of normal dermal appendages such as hair follicles and sebaceous or sweat glands in the connective tissue. Teratoma - Dermal appendages associated with mesodermal and endodermal derivatives such as osseous and dental structures and respiratory and intestinal epithelium. The term "Dermoid" was suggested as a clinical term for all developmental cysts of the floor of the mouth (Meyer 1955). A possible cause o f those formations is entrapment of epithelial debris or rests during the midline closure of the first and second branchial arches. Some of the cysts may be formed from remnants of the "tuberculum impar of His" which during the third and fourth week of intrauterine development will form, together with the inner surface of each mandibular arch, the body of the tongue and the floor of the mouth. Another possibility is the submergence of ecto-
213
dermal cells at the time of closure of the anterior neuropore. In the literature from 1949-1967 reviewed by Y o s h i m u r a et al. (1970) only 7 congenital cysts were included among reported cases. Clinically it is important to establish the relation of the cyst to the mylo-hyoid and genio-hyoid muscles which make the swelling appear in a sublingual or submental position. This determines whether the approach to the tumour should be intraorally or through an external incision. Sometimes a combined approach is necessary. The differential diagnosis is: Ranula (originating from Wharton's duct); thyroglossal duct cyst; cystic hygroma; branchial cleft cyst; acute infection or cellulitis of the submaxillary and sublingual glands or floor of the mouth; tumours of salivary glands; normal fat and lipomata. Conclusion
Dermoid cysts of the floor of the mouth are rare. The most interesting feature of the case presented is the difference in epithelial lining between the closely linked cystic structures. A difference in development can therefore be suspected.
References Gold, B. D., D. E. Sheinkop[, B. Levy: Dermoid, epi-
dermoid and teratomatous cysts of the floor of the mouth. J. oral Surg. 32 (1974) 2 Hoggins, G. S.: Congenital sublingual cystic swelling due to imperforate salivary duct. Oral Surg. 37 (1974) 370 Hooley, ]. R., G. L. Freedman, R, S. Redma,nn: Sublingual dermoid cyst in an infant. Oral Surg. 24 (1967) 715 Meyer, ],: Dermoid cysts of the floor of the mouth. Oral Surg. 8 (1955) 1149
New, G. B., J. B. Erich: Dermoid cysts of the head
and neck. Surg. Gynec. Obstet. 65 (1937) 48 Yoshimura, Y., K. Takada, M. Takeda, T. Mimura, M. Mori: Congenital dermoid cysts of the sub-
lingual region. Report of case. J. oral Surg. 28 (1970) 366 M. R. I4Zexler, M.D., Plastic Surgery, H.U.H. P.O. Box 499, ]erusalem/Israel