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Dermoid cysts (dermoids) of the floor of the mouth
Oral DERMOID
CYSTS
Pathology
(DERMOIDS)
OF THE
FLOOR
OF THE
MOUTH
Introduction ERMOID cysts (dermoids) of the floor of the mouth are rare; four cases are reported in this art.icle. New and Erich’. 2 report.ed that of 1,495 dermoid cysts seen at the &layo Clinic from 1910 to 1935, only 103 (6.94 per cent) were in the head and neck and, of these, twent.y-four were in the floor of the mouth. Shore3 found four cases of dermoid cyst,s of the floor of tht mouth in 54,000 surgical specimens. Meister,’ in 1937, collected a total of 143 from the world literature, of which many of the earlier cases were not proved histologically. Approximately fifteen dermoids of the floor of the mouth ha.ve been added since 1937, to increase the total number of report,ed cases t,o less t,han 160 in a1L3-16 These cysts are equally distributed in both male and female. h’ew and Erich’, * report that 51 per cent were in males and 49 per cent in females; Meister W statistics are essentia.lly the same-54 per cent‘ males and 46 per cent females. Of the four cases reported in this paper, three were in males. and one in a female. There is great variation in reported age of occurrence of dermoitls of the floor of the mouth; the average lies between 15 and 35 years of age. N’hcreas New and Erich’, z Ii reported a dertnoid in a 72-year-old man. one of t.he C:ISW Alt,hough they ma>- Ilot lje presented in this art,icle was present at birth. manifest until many years later, these cysts are usually regarded as congrnital in na.ture.18 All dermoids of the floor of the mouth have basica.lly the same clinical appearance j however, there is considerable variation in the microscopic pict.ure. Therefore, a new classification based upon the histopat.hologic findings of these cysts will be present,ed and discussed.
D
Embryology
and Development
Historically, in 1859 Roser first described them as epidermoid tumors; he was supported by Virchow, &hide, and others. Hulkc (1862) incorrectly From the Department of Oral Pathology and Oral Surgery, Tufts Dental School. Presented at the ninth annual meetinn of the American Academy of Oral Pathology, Chicago, Illinois, Feb. 6 and 7, 1955.
1150
IRVING
MEYER
considered them to be atheromatous cysts of the tongue. Chiari (1891) considered them congenital tumors resulting from the emhryonal closing off of epidermal cells. In 1878 Mikulicz’8 stated that dermoids of the floor of the mouth c~ultl occur in three different ways, namely : (1) t,hrough midline closures of body cavities ; (2) through closure of channels or clefts which, during fetal life, were covered with epithelium ; and (3) through abnormal deposits of epidermis ill deeper tissues. The majoriby of dermoids are deveIopmenta,l cysts derived from epithelial debris or rests enclaved during the midline closure of the bilateral mandibular (first) and hyoid (second) branchial arches. Some of these cysts may be formed by remnants of the t,uberculum impar of His which, togethel with the lateral processes from the inner surface of each mandibular arch. form the body of the tongue and floor of the mouth. These developments take place during the third and fourth weeks of embryonic life.+ 18-Z] .It has further been suggested that some of the cells enclaved during these fusion processes are totipotent blastomeres and, therefore, derivatives of any or all of the three basic germ layers may be present in dcrmoids.20. 21 This was probably the thought that led Ehrich2*, 23 to employ the term dysontogenesis, meaning “ disturbance in development, ” in referring to dermoids of the floor of the mouth. The growth of these cysts, either gradual or sudden as the case may be. is not clearly understood. Verneuil and Cladols stated that sudden growth was due t,o bact,erial infection, but ca,reful bacteriologic studies have shown t,hat many of these cysts contain sterile material. Most authors suggest that the development of these cysts occurs during the period of increased activity of the epithelial tissues, such as sweat glands, hair, etc.. continually filling t,he lumen of the cyst. This increased growth activity coincides with the ages of 15 to 35 years, when most of these lesions occur.
Classification Various classifications of dermoids of the floor of the mouth have been suggested, but none are completely satisfactory. Most are based on purely anatomic position of the cysts in relation to (a) the musculature and (b) the mandible and hyoid bone. Ilarker, in 1883, classified dermoids of the floor of the mouth on the basis of their location only; thus : (a) Median cyst (midline) (b) Lateral cyst 1. Unilateral 2. Bilateral The bilateral type has ncvcr been rcportetl, but is theoretically possible by a division of a median cyst by a connection of the median raphe of mylohyoid muscle below with the frenum of the tongue above.4y *a Willinger,4 in 1918, described a dermoid cyst which was divided in two by the mylohyoid, that is, superior and inferior daughter cysts.
DERMOID
7151
CYSTS
In 1886 Marchant, studying thirt.y collected cases, classified them by their attachments to either the mandible, as “kystes dermoid ad geniens,” or to hyoid bone, as “kystes dermoid ad hyoid.” This classification is not valid, inasmuch as most dermoids of the floor of the mout,h do not attach to either bone.ls Of the four cases presented here, one attached “ad geniens,” one “ad hyoid, ’ ’ and the other two were not attached to either bone. Dehonnelle (1908), on twenty-five collected cases, related his classification to the position of the cysts to the adjacent musculature, these cysts being between mylohyoid muscle and oral mucous membrane.4’ 14,I*, 24,25 This classification, which is quite useful to the surgeon, is as follows (Fig. 1) : (A)
Median 1. Below geniohyoid 2. Above geniohyoid
(submental (sublingual
position). position).
(B) Lateral (rare) These would be located on either side in the space formed by the medial surface of mandible laterally, the oral mucous membrane superiorly, the tongue musculature medially, and the mylohyoid This type might be a median cyst which has shifted inferiorly. laterally into the space thus described.
SUftCIN6UAL SUBMENTAL
Fig.
l.-Diagram
illustrating
the
DERMOID two
CYSTS -
positions which OCCUpY.
dermoid
cysts
of
floor
of
mouth
may
Using the terms dysontogenetic,**v 23 epidermoid,4* I7 dermoid,17s 26*27 teratoid, or teratoma,*lp 23126 and the germ layer hypothesis of development of these cysts as suggested by various authors, including Ewing,*l Boyd,20 C01p,~~ etc., I propose the following classification on histopathologic considerations of these cysts of the floor of the mouth: Dysontogenetic Cysts of the Ploor of the Mouth (a) Epidermoid (b) Dermoid (c) Teratoid
1152
IRVTNG
MEYER
(a) Epirlermoicl cyst of fioo~ of nlowth: An epithelial-lined rounded by a. capsule with no skin appendages prrsent.
cavity sur-
An epithelial-lined cavit,y with (1)) IIeYrGtl cyst of jtoor, of’ north: skin appendages of hair, hair fol liclcs, scbaceoux glands, sweat glands, etc., present in t,he underlyin g eonn&ive tissue. This is a compound cyst. cavity with (c) Terntoid cyst of floor of mouth: An epithelial-lined the following elements present in the capsule: (1) skin appendages including hair follicles, sebaceous glands, sweat glands, keratin, etc. ; (2) connective tissue derivatives such as fibers, bone, and gastroinnmscle, blood vessels, etc. ; and (3) respiratory testinal tissues. This is a complex cyst. Because of its long usage and presence in the literature, dermoid should be retained as a clinical terrn for all types of dysontogenetic or developmental cyst,s of the floor of the mouth. The acquired traumatic implantation cyst of the floor of the mouth is excluded from this proposetl classification.
Clinical Aspects and Differential
Diagnosis
Clinically, dcrmoids of the floor of the mouth are frequently quite striking in their appearance. Those presenting themselves intraorally, or suhlingually, may actually displace the tonguc upward to the palate until difficulty in eating, speaking, and even breathing may occur; those presenting extraorally, or submentally, usually appea,r as a pendulous mass beneath the feel, but may feel cystic, mandible. The cysts generally have :I “doughlike” depending on consistency of the contents, which ma.y vary from a cheesy, Hair, nails, keratin, sebaceous-like substance to n more liquefietl material. etc., may be present,28 depending on the type of cyst being dealt wit,h. Some have frank pus present due t,o arute infection. These lesions vary in weight from one gram to several hundrccl grams, a,nd may va.ry in size frorn a small pea-sized growth to one the size of a large grapefruit. Sinus tracts may develop from these cysts to open either intraorally into the floor of the mouth or extraorally into t,he skin beneath the chin. Dermoids may undergo malignant degeneration and may metastasize to lymph nodes.‘, Ii, 20,26 The differential diagnoses of tlermoids of the floor of the mouth include: (I) ranula, (2) unilateral or bilateral blockage of Wharton’s ducts, (3) thyroglossal duct cyst. (4) cystic hygroma,, (5) branchial cleft cysts, (6) acute infection or cellulitis of the floor of the mouth, (7) infections of submaxillary and sublingual glands, (8) benign and malignant tumors of the floor of the mouth and adjacent salivary glands. and (9) normal fat mass in the submental area. The calinical diagnosis is inconclusive.
Treatment The treatment of dermoid cysts of the floor of the mouth is surgical. Aspiration may be used for temporary relief. Aspiration and sclerosing agents, as definitive therapy, are not good; these may lead to even more difficulties, including
DERMOID
1153
CYSTS
acute infection and ccllulitis of the surrounding area. There have been no reports of the use of radiation, but it can readily be assumed that this would not be effective. The surgical approach can he made either intra- or extraorally, depending on the position of the cyst in relation to t,he mylohyoid and geniohyoid muscles. Those lying between geniohyoid and oral mucous membrane (sublingual position) are best removed by an intraoral approach ; those lying between geniohyoid and mylohyoid (submental position) are approached through a transverse incision t,hrough skin, fascia, platysma, and mylohyoid. Usually the cysts are readily shelled out by blunt dissection, with difficulty being encountered only if there has been mattin g down due to previous treatment by aspiration or sclerosing solutions or episotles of infection. Case Reports white married man, Case l.*-Feb. 4, 1954. The patient, S. K., was a 33-year-old which first appeared three years whose chief complaint was “swelling of the throat” earlier. At that time his local physician incised ant1 drained a tender “golf ball-sized” mass intraorally, and a large quantity of whitish, nonfoul material exudetl. The mass did
Fig.
‘.-Case
1.
Preoperative
v&v
showing
submental
mass.
not recur, but the patient went to a clinic for further advice; nothing was found on examination. About ten months later, the mass gradually recurred and increased in size to 8 by 3 cm. Upon bimanual palpation, a doughlike mass could be felt. The mass did not readily displace itself intraorally and appeared to be somewhat more accessible through a skin approach. The family history and laboratory studies were normal. Submandibular dermoid. Preoperative diagnosis: Surgeon : Assistant
Dr. Fred A. Post, Springfield, surgeon:
Date of surgery: *Courtesy
Dr. Irving
Massachusetts.
Meyer.
Feb. 6, 1954.
of Dr. 8’. A. Post, Lesson
Memorial
Hospital,
Springfield,
Massachusetts.
1154
IRVING
MEYER
Fig. 4. Fig. I.-Case 1. Photomicrograph (IOMV-power) of cyst wall showing epithelium. piling up of keratin into the lumen, and chronic inflammation. Fig. 4.-Case 1. Photomicrograph of cyst ~~11 (high-porei”).
stratii%
srluamous
cyst was easily freed by blunt finger dissection and extirpated in toto. The geniohgoid, mylohyoid, and platysma were sutured and a rubber dam drain m-as placed to the depths of the cavity. The skin was then closet1 with black silk. (In retrospect, both surgeons felt that an intraoral approach would have been better than the extraoral approach used.) The patient hat1 an uneventful WCO~~ZI’~ RII(~ has been examined periodically with no evidence of recurrence of the cys:t. Pathology
rc,port:
Gross description: ‘l’hc spwiruen consisted of an oval mass of tissue measuring 5 by 3 by 3 em., the surface of which was generally smooth with small collections of shaggy fibrous tissue present at various points of dissection. The
specimen was generally yellowish pink in color, soft in consistency, and cystic in nature. The mass weighed 29 grams and, upon sectioning, was filled with a cheeselike material. Microscopic aPs~i’iptio~a: Sections consistetl of stratified squamous-cell epithelium with considerable piling of keratin into the lumen. The untlerlying connective tissue was dense and containetl l~loocl vessels, fibrous tissue, and inflammatory cells. Cholesterol slits were present in the connective tissue. Epidermoitl cyst of the floor of the mouth; no evi,I~~CVOSCO~~C aiapt0sis : dence of malignancy. Case 2.*-On Sept. 2, 1951, A. (1., a &Z-year-old unmarried white woman, presented a 10 cm. pendulous mass hanging beneath the mandible and apparently adherent bilaterally The skin was freely movable over the to inner surfaces of the angles of the mandible. mass, which could not be presented intraorally upon submental digital pressure. There was a history of swelling in this area having been incised and tlrained when the patient until the age of 15, when the preswas 2 years oltl. The patient remained free of complaint it, gradually increased to its present size. There was ent mass became very evident;
Fig. 5.-Case ueither patient weight.
2.
Lateral
view
showing
submental
mass
The pain nor discomfort, but the patient was quite conscious of its unsightliness. had been placed on a low-fat, high-protein diet by her physician in order to lose The family history and laboratory findings were normal.
Preoperative suryeon: Assistant
dictptosis:
Dr. Arthur su,rgeon :
Date of swgmy:
Dermoid
cyst of the floor of the mouth.
E. Qramse, Hpriugficld, Dr. Irviug
Massachusetts.
Xicycr.
Sept. 7, 1951.
gcueral anesthesia, with nasal intubation, a high Pindings and procedure : Vndrr transverse incision was matle in the upper neck through skin, subcutaneous tissues, and platysma, ant1 was extencletl back to the angles of the mandible on each side. The mplohyoid muscles were split transversely following the outline11 iucisiou. The anterior belly By blunt dissection, the of each digastrie muscle was exposetl and retracted laterally. present top of the cystic mass was exposed; there was evidence of previous inflammation *Lesson
Memorial
Hospital,
SpringfIeld,
Massachusetts.
1156
1RVING MEYER
1)~ the marked adherence of the mylohyoid muscles to the inferior aspect of the cyst, ant1 geniohyoid muscles to the superior aspect of the cyst. The carotid sheaths of both sides were carefully freed up from the field and retracted. The cyst was then completely freed up from its firm adhesions to the angles of the mandible, base of the tongue, and hyoid bone; it was firmly adherent to and enveloped at the trachea somewhat on each side. The cyst was finally deliverecl intact through the incision without compromising any of the larger nerves or vessels of the neck. The geniohyoitl and mylohyoid muscles, the deep cervical fascia, platysma, and subcutaneous tissues were approximated with catgut suture. The wound was drainetl with a strip of rubber dam, ant1 the skin was closed 1)~ a plastic technique with black silk suture.
Fig.
G.-Case
2. -. Photonlicrograph uell lining, keratin,
(low-power) am1 a hair
The patient had an uneventful postoperative tions have shown no recurrence of the lesion. Pathology
showing cyst wall with stratifled follicle cut in cross section. course and repeatetl
follow-up
squamous examina-
report:
Gross description: The specimen consisted of a large cyst measuring 10 by 5 by 5 em. and containing a granular cheesy material. The cyst wall was tra.nslucent and measured 0.5 mm. in thickness. It presented a smooth inner surface and a roughened outer surface. The specimen weighed 250 grams. AratiHioroscopia aesO~iptiOl~: Hec:t,ions eonwistrd of a st,rip of krratiniertl fied squamous epithelium bortlrred by dense connective tissue containing an occasional chronic: inflammatory ~11. In one area, sevt~ral hair follices were cut in transverse section . L L .,, Microscopic diagnosis: Cyst wall with skin appendages, cal diagnosis of dermoid cyst; no evidence of malignancy. Case 3.*-Aug. 14, 1953. The patient, whose chief complaint was a swelling in the speech. Inasmuch as he was a salesman, this had been given a diet by an internist to help
consistent
with rlini-
H. R., was a 30.year-old married white man floor of the mouth which interfered with his had become a serious problem and the patient him lose weight in this area.
Examination demonstrated a massive swelling in the floor of the mouth which had so raised the body of the tongue palatally that only about 2 cm. of the tip of the tongue The mass could be displaced upward into the mouth by extraoral digital was evident. *Wesson
Memorial
Hospital,
Springfleld,
Massachusetts.
DERMOID
CYSTS
1157
pressure in the submental area. The carunculae of Wharton’s determined. General physical examination and laboratory studies history was noncontributory. Preoperative Surgeon : Assistant
diagnosis: Dr. Irving surgeon
Dermoid
cyst of the floor of the mouth.
Meyer. Dr. Lawrence
:
ducts could scarcely be were normal; the family
Van Selden, Springfield,
Massachusetts.
28, 1953. a traction Findings oxd procedure : Under general anesthesia, with nasal intubation, suture was placed through the body of the anterior third of the tongue. A midline vertical incision was started at the tip of the tongue on the veutrum and carried downward through Date
of surgery:
Aug.
Fig.
Fig.
S.-Case
3.
‘I.--Case
Intraoral
3.
Photograph
photograph
showing
showing
mass
submental
displacing
mass.
the
tongue
to the
palate.
the mucous membrane over;ying the mass for a distance of .I cm. The incision was then extended between the carunculae of Wharton’s ducts to the lingual plate of the alveolus of the mandible. Using blunt dissection, the oral mucous membrane was freed up bilaterally to expose a shiny, silver-white mass beneath. The tlucts of Wharton were identified and retracted from the fielll. The dissection was carried deep around the entire mass, which was approximately the size of a grapefruit. After considerable blunt finger and hemostat dissection, the mass was completely freed from the surrounding musculature and fascia, and was found to be attached b,v a pedicle to the genial tuhercles of the lingual
Vi%. II). Fig. 9.- -(:asc ! 3. weigl zing 325 3m. Fig. 1 o.- -Ck 3e 3. chole stem A-lik e Ins lterial.
Fig.
Il.--Case
3.
Photograph Surgical
Photomicrograyh epitheliunr,
of
surgical
specimen
cut
specimen
measuring
open
to show
(medium-power) hair follicle, an,i
showing sebaceous
contents
a lining g-lnn~ls.
8 by
6
consie iti
Of stratified
5
.‘“I.
of a cht
S~lUarnoUS
DERMOID
CYSTS
1159
aspect of the symphysis of the mandible. The petlicle was clamped between two clamps, cut, and tied. Because of its great size, the mass was tleiivered with some difficulty from the mouth without rupture. The geniohyoitl muscles lying above much stretched mylohyoid muscles were reatlily identified at the base of the large residual spare. The lingual nerves and vessels, as well as genioglossal musculature, were also i(lentifiet1. The lateral walls of the space were brought together with catgut and the oral mucoos membrane was closed with black silk suture. The patient
(lid well postoperatively
and has hall no recurrence
of the lesion.
Gross descripticm.: The specimen consisted of a large cystic structure measuring 8 by 6 by 5 cm. On cut section, the lumen was filled with a cheesy white material; the inner lining was smooth and glistening throughout. The mass weighed 325 grams. It was covered with fine blood vessels on its outer surface. xi~~~.~~pi~ ae.kpti07~ : Sections showed an elongated strip of tissue consisting of dense fibrous connective tissue covered with stratified squamous epiThe epithelium was composed of the different layers from stratum thelium. germinativum to stratum eorneum. There was a notable hpperkeratosis but Hair follicles, some plugged with keratin, were the stratum spinosum was thin. in evidence; sebaceous glands were present in the connective tissue. clinical
NLlicroscopio diagnosis: diagnosis of dermoid
Cyst wall with skin appendages, consistent with There was no evidence of malignancy. cyst.
the
Case 4.x:--April 12 7 1950 . The patient, F. I:., vvab a 2-day-old male Negro infant who, according to the obstetrical service, was born with “two tongues.” The inferior “tongue” which was, on examination, a sublingual, doughlike mass measuring L’ cm. in circumference, The mass was aspirated and 2 C.C. of milky was causing some respiratory embarrassment. The patient was taken home by t,he parents against advice. white fluill was obtained.
Fig.
12.-Case
4.
Clinical
photograph
showing
mass
displacing
tongue
The mass recurred and was again aspiratetl in March, 1950. Bacteriologic aspirations were essentially negative. In April, 1950, the mass recurred brought the infant in for surgical removal of the growth.
upward.
studies of both and the mother
Clinical examination demonstrated a midline saelling beneath the tongue at the base of the lingual frenum. It measured about 2 by 1 cm. on its oral surface and caused a marked upward displacement of thy! tongue. General physical examination and laboratory studies were normal; the family history was noncontributory. Preoperative
Surgeon : Fred
diagnosis: Dr. Irving
*Department of Lucchesi. Chief.
Dermoid
cyst in sublingual
space.
keyever.
Dentistry,
Philadelphia
General
Hospital,
Philadelphia,
Pennsylvania,
1160
IRVING .dSSistant
1)~. Thomas
.Wr{JeO’rls:
Bate of surgery:
April
Meloy
MEYER and Milton
Ivkcr,
Philadelphia,
Pennsylvania.
14, 1950.
Findings and pnxcdwre: Gntler general anesthesia, with nasal intubation, a retraction suture was placed t,hrough the anterior portion of the tongue. B midline incision was started on the ventrurrl of tongue I cm. from the top and carried downward over the protrusion of the cyst between the bilateral carunculae of Wharton’s ducts. The incision was
Fig. 13.-case
4.
Surgical
specimen
measuring
3 by 2 by 2 cm.
Fig. Id.-Case 4. A very low-power view of the cyst wail showing muscle bundles in longi and cross section, lining of the lumen with epithelium, and a connective tissue Capsule surrounding the entire cyst. spread with blunt dissection and carried downward until the body of the clearly exposed. There was a distinct fascial separation between the The geniohyoid and rounding musculature and it was delivered intact. The space and oral at the base of the remaining space were identified. were closed with catgut sutures. The patient made an uneventful recovery and t,here has been no lesion.
cystlike mass was mass and the surmylohyoid muscles mucous membrane recurrence
of the
DERMOID
Pathology
CYSTS
1161
report:
The specimen consisted of a mass measuring 2 by 1 by Gross description: 1 em., encapsulated by a brown fibrous tissue and weighing 3 grams. Upon sectioning the specimen, the cut surface was white and glistenin,g. There was a central lumen, and many small white appendages werr present very slightly raised from the surface. The content of the lumen was a cheesy white material. Fig.
Fig.
15.
16.
Fig. 15.--Case 4. Photomicrograph (medium-power) showing stratified sguamous epitheli urn, hair follicles, sebaceous glands, and keratin in the lumen. l?ig. l&-Case 4. Photomicrograph (medium-power) showing the cyst wall wit .h a *.?A?”ldostratifled columnar epithelial lining, sudoriferous glands, an arteriole, fat tissue, and flbrc bus connective tissue.
1162
IRVING
MEYER
Microscopic desoiptiou : Swt~ions of thv sprc~inlvn ~howetl ali elougat,ed, large lumen lined with a ~callupeci tmlder of it stratifietl squamous epithelium. The epithelium varied in thicknes* from ten to three or four cells, and W:H seen to vary in regions-areas of keratin forulation. pronounced granulosum veils. and cuboidal basal cells which :IW contrastetl tjy lack of keratinization.
Fig.
18.
(me~lium-pobver) showing lining of stratifled e Fig. l’i.-Case 4. Photomicrograph sebaceous glands, and keratin shedding us epithelium, hair follicle in cross section, 1unnen. Fig. 18.-&w 4. Photomicrograph (high-pov’er) of cyst wall (lemonstrating the 11 rolunmar epithelium (nasal type), skeletal muscle. and vas( of ciliated pseudostratified chr mnels.
Portions of the lining Svere comp,o.serl of l~se~~~lo~t~tltifie~l~ colunlnal~, ciliate11 epithelium typical of respiratory (nasal) epitheliun~. The epithelium was not clearly (lelineatetl, iu its entirety, from the uuderThe connective tissue varied lying connective tissue b)- it basement membrane. and contained areas of smooth muscle fibers. in its cellularity and vascularit?;
DERMOID
CYSTS
1163
Comment The four CRWSof cysts of the floor of the mouth presented lend themselves to the classifica.tion suggested in t,his article : Dysontogenetic Cysts of the Floor of the Mouth (a) Epidermoid-Case 1. (b) Dermoid-Cases 2 and 3. (c) Teratoid-Case -I. Case 1 is that of a simple stratified squamous epithelial-lined cyst WIrounded by a connective tissue capsule; this is the epidermoid cyst wit,h its lumen filled with a caseous material composed in part of cholesterol. Cases 2 and 3 are those of stratified squamous epithelial-lined cysts with skin appendages, including hair follicles, sebaceous glands, and sweat glands present in the connective tissue capsule. These are the classic dermoid cysts of the floor of the mouth. Case 4 is that of a teratoid cyst because of the complexity of its conelements are present : (1) epithelial tissues stituent tissues. The following consisting of stratified squamous epithelium and skin appendages, including hair follicles and sebaceous and sudoriferous glands; (2) connective tissues, including the usual vascular and fibrous elements present, striated muscle in both thick bundles and isolated fibers, and large distinct areas of fat tissue; and (3) pseudostratified, columnar, ciliated epithelium of respiratory origin. Summary Dermoid cysts of the floor of the mouth are discussed as to their embryolA classification of these cysts, based ogy, development, and classification. on histopathologic findings, is presented in this article and it is suggested that the word dermoirl be retained as a clinical term for all these developmental cysts of the floor of the mouth. Four cases of dermoid cysts of the floor of the mouth are presented; these were successfully managed surgically without recurrence. A preoperative eva,luation of the location of the cyst in relation to the mylohyoid and geniohyoid muscles, that is, sublingual or submental position, is important in determining whether t.o approach the cyst through an intraoral or an extraoral incision, The clinical diagnosis is inconclusive and the final diagnosis must bc proved by histologic examination.
1164
IRVISG
MEYER
References 1. New, G. B.: Congenital Cysts of the Tongue, the Floor of the Mouth, the Pharynx, and the Larynx, Arch. Otolaryng. 45: 145-158, 1947. 2. New, G. B., and Erich, J. B.: Dermoid Cysts of the Head and Neck, Surg., Gynec. & Obst. 65: 48-55, 1937. 3. Shore, B. R.: Sublingual Epidermoid Cysts, Ann. Surg. 108: 305-308, 1938. 4. Johnston, W. H.: Cysts of the Floor of the Mouth, Ann. Otol., Rhin. & Laryng. 51: 917-930, 1942. 5. Bernier, J. L., and Ash, J. E.: Atlas of Dental and Oral Pathology, Washington, 1948, Registry Press. 6. Cook, T. J.: Dermoid Cyst. A Report of a Case, ORAL SURG., ORAL MED., AND ORAL PATH. 3: 740-742, 1950. Cystic Teratoma of the Head, Plast. & Reconstruct. 7. Curtis, L., and Ecker, H. A.: Surg. 3: 603-612, 1948. 8. Figi, F. A., and Dix, C. R.: Dermoid Cysts of the Floor of the Mouth. Report of a Case, Proc. Staff Meet., Mayo Clin. 14: 289-293, 1939. 9. . Frenette, O., and Ha114 J.: Sublingual Cyst: Surgical Therapy of a Case, Lava1 m&l. 9: 708-710, 1944. Two Cases of Sublingual Dermoid (Cyst), Am. J. Ortho10. Ivy, R. H., and Curtis, L.: dontics and Oral Surg. 31: 666, 1945. 11. Kini, M. G.: Sublingual Dermoids, Indian J. Surg. 1: 25-27, 1939. Leo: Dermoid Cyst, of the Floor of the Mouth, U. S. Armed Forces M. J. 2: 12. Korchin, 288-292, 1951. Epidermoid Cysts, Arch. Otolaryng. 33: 659-662, 1941. 13. Marshall, L. R.: Sublingual 14. Masuda, B. J.: Dermoid Cyst in the Floor of the Mouth, Am. J. Orthodontics and Oral Surg. 32: 252-256, 1946. Dermoid Cyst, Canad. M. A. J. 63: 480, 1950. 15. Musgrove, J. E.: Sublingual Clinical Pathology of the Tongue, ORAL SURG, ORAL MED., AND ORAL 16. Schaffer, Julius: PATH. 5: Si-111, 1952. Sebaceous, Mucous, Dermoid and Epidermoid Cysts, Am. J. Surg. 60: 17. Erich, J. B.: 672, 1940. Dermoid Cysts of the Floor of the Mouth, Surg., Gynec. & Obst. 40: 183. 18. Colp, R.: 195, 1925. Oral Histology ant1 Embryology, ed. 2, St. Louis, 1949, The C. V. Mosby 19. Orban, B.: Company. A Textbook of Pathology, ed. 4, Philadelphia, ‘1944, Lea & Febiger, p. 308. 20. Boyd, Wm.: Diseases, ed. 4, Philadelphia, 1940, W. B. Saunders Company. 21. Ewing, J.: Neoplastic Philadelphia, 1941, Lea & Febiger. Ehrich, W. E.: Pathology, Parasites of the Mouth, Am. J. Orthodontics and Oral Surg. ii: Ehrich, W. E.: Teratoid 31: 650-659, 1945. Oral Pathology, ed. 3, St. Louis, 1954, The C. V. Mosby Company, 24. Thoma, Kurt H.: p. 835-837. Dermoids, Lancet 1: 297, 1930. 25. Warren, R., and Cooper, N. C.: Sublingual Pathology of Tumours, London, 1948, Butterworth & CO., Ltd. 26. Willis, R. A.: Ollerenshaw, R.: On Dermoids of the Tongue, Brit. M. J. 1: 1290-1291, 1912. Kronfeld’s Histopathology of the Teeth and Their Surrounding Struc22;: Boyle, P. E.: tures, ed. 3, Revised, Philadelphia, 1949, Lea & Febiger. Dermoid Cyst of the Floor of the Mouth (Case 29. Cameron, J. R., and Boyko, G. V.: Report), J. A. M. A. 89: 1149, 1927. 30. Stein, 0. J.: Cysts in Floor of Mouth, Ann. Otol., Rhin. & Larpng. 34: 1028, 1925.
CYSTS
Pathology
(DERMOIDS)
OF THE
FLOOR
OF THE
MOUTH
Introduction ERMOID cysts (dermoids) of the floor of the mouth are rare; four cases are reported in this art.icle. New and Erich’. 2 report.ed that of 1,495 dermoid cysts seen at the &layo Clinic from 1910 to 1935, only 103 (6.94 per cent) were in the head and neck and, of these, twent.y-four were in the floor of the mouth. Shore3 found four cases of dermoid cyst,s of the floor of tht mouth in 54,000 surgical specimens. Meister,’ in 1937, collected a total of 143 from the world literature, of which many of the earlier cases were not proved histologically. Approximately fifteen dermoids of the floor of the mouth ha.ve been added since 1937, to increase the total number of report,ed cases t,o less t,han 160 in a1L3-16 These cysts are equally distributed in both male and female. h’ew and Erich’, * report that 51 per cent were in males and 49 per cent in females; Meister W statistics are essentia.lly the same-54 per cent‘ males and 46 per cent females. Of the four cases reported in this paper, three were in males. and one in a female. There is great variation in reported age of occurrence of dermoitls of the floor of the mouth; the average lies between 15 and 35 years of age. N’hcreas New and Erich’, z Ii reported a dertnoid in a 72-year-old man. one of t.he C:ISW Alt,hough they ma>- Ilot lje presented in this art,icle was present at birth. manifest until many years later, these cysts are usually regarded as congrnital in na.ture.18 All dermoids of the floor of the mouth have basica.lly the same clinical appearance j however, there is considerable variation in the microscopic pict.ure. Therefore, a new classification based upon the histopat.hologic findings of these cysts will be present,ed and discussed.
D
Embryology
and Development
Historically, in 1859 Roser first described them as epidermoid tumors; he was supported by Virchow, &hide, and others. Hulkc (1862) incorrectly From the Department of Oral Pathology and Oral Surgery, Tufts Dental School. Presented at the ninth annual meetinn of the American Academy of Oral Pathology, Chicago, Illinois, Feb. 6 and 7, 1955.
1150
IRVING
MEYER
considered them to be atheromatous cysts of the tongue. Chiari (1891) considered them congenital tumors resulting from the emhryonal closing off of epidermal cells. In 1878 Mikulicz’8 stated that dermoids of the floor of the mouth c~ultl occur in three different ways, namely : (1) t,hrough midline closures of body cavities ; (2) through closure of channels or clefts which, during fetal life, were covered with epithelium ; and (3) through abnormal deposits of epidermis ill deeper tissues. The majoriby of dermoids are deveIopmenta,l cysts derived from epithelial debris or rests enclaved during the midline closure of the bilateral mandibular (first) and hyoid (second) branchial arches. Some of these cysts may be formed by remnants of the t,uberculum impar of His which, togethel with the lateral processes from the inner surface of each mandibular arch. form the body of the tongue and floor of the mouth. These developments take place during the third and fourth weeks of embryonic life.+ 18-Z] .It has further been suggested that some of the cells enclaved during these fusion processes are totipotent blastomeres and, therefore, derivatives of any or all of the three basic germ layers may be present in dcrmoids.20. 21 This was probably the thought that led Ehrich2*, 23 to employ the term dysontogenesis, meaning “ disturbance in development, ” in referring to dermoids of the floor of the mouth. The growth of these cysts, either gradual or sudden as the case may be. is not clearly understood. Verneuil and Cladols stated that sudden growth was due t,o bact,erial infection, but ca,reful bacteriologic studies have shown t,hat many of these cysts contain sterile material. Most authors suggest that the development of these cysts occurs during the period of increased activity of the epithelial tissues, such as sweat glands, hair, etc.. continually filling t,he lumen of the cyst. This increased growth activity coincides with the ages of 15 to 35 years, when most of these lesions occur.
Classification Various classifications of dermoids of the floor of the mouth have been suggested, but none are completely satisfactory. Most are based on purely anatomic position of the cysts in relation to (a) the musculature and (b) the mandible and hyoid bone. Ilarker, in 1883, classified dermoids of the floor of the mouth on the basis of their location only; thus : (a) Median cyst (midline) (b) Lateral cyst 1. Unilateral 2. Bilateral The bilateral type has ncvcr been rcportetl, but is theoretically possible by a division of a median cyst by a connection of the median raphe of mylohyoid muscle below with the frenum of the tongue above.4y *a Willinger,4 in 1918, described a dermoid cyst which was divided in two by the mylohyoid, that is, superior and inferior daughter cysts.
DERMOID
7151
CYSTS
In 1886 Marchant, studying thirt.y collected cases, classified them by their attachments to either the mandible, as “kystes dermoid ad geniens,” or to hyoid bone, as “kystes dermoid ad hyoid.” This classification is not valid, inasmuch as most dermoids of the floor of the mout,h do not attach to either bone.ls Of the four cases presented here, one attached “ad geniens,” one “ad hyoid, ’ ’ and the other two were not attached to either bone. Dehonnelle (1908), on twenty-five collected cases, related his classification to the position of the cysts to the adjacent musculature, these cysts being between mylohyoid muscle and oral mucous membrane.4’ 14,I*, 24,25 This classification, which is quite useful to the surgeon, is as follows (Fig. 1) : (A)
Median 1. Below geniohyoid 2. Above geniohyoid
(submental (sublingual
position). position).
(B) Lateral (rare) These would be located on either side in the space formed by the medial surface of mandible laterally, the oral mucous membrane superiorly, the tongue musculature medially, and the mylohyoid This type might be a median cyst which has shifted inferiorly. laterally into the space thus described.
SUftCIN6UAL SUBMENTAL
Fig.
l.-Diagram
illustrating
the
DERMOID two
CYSTS -
positions which OCCUpY.
dermoid
cysts
of
floor
of
mouth
may
Using the terms dysontogenetic,**v 23 epidermoid,4* I7 dermoid,17s 26*27 teratoid, or teratoma,*lp 23126 and the germ layer hypothesis of development of these cysts as suggested by various authors, including Ewing,*l Boyd,20 C01p,~~ etc., I propose the following classification on histopathologic considerations of these cysts of the floor of the mouth: Dysontogenetic Cysts of the Ploor of the Mouth (a) Epidermoid (b) Dermoid (c) Teratoid
1152
IRVTNG
MEYER
(a) Epirlermoicl cyst of fioo~ of nlowth: An epithelial-lined rounded by a. capsule with no skin appendages prrsent.
cavity sur-
An epithelial-lined cavit,y with (1)) IIeYrGtl cyst of jtoor, of’ north: skin appendages of hair, hair fol liclcs, scbaceoux glands, sweat glands, etc., present in t,he underlyin g eonn&ive tissue. This is a compound cyst. cavity with (c) Terntoid cyst of floor of mouth: An epithelial-lined the following elements present in the capsule: (1) skin appendages including hair follicles, sebaceous glands, sweat glands, keratin, etc. ; (2) connective tissue derivatives such as fibers, bone, and gastroinnmscle, blood vessels, etc. ; and (3) respiratory testinal tissues. This is a complex cyst. Because of its long usage and presence in the literature, dermoid should be retained as a clinical terrn for all types of dysontogenetic or developmental cyst,s of the floor of the mouth. The acquired traumatic implantation cyst of the floor of the mouth is excluded from this proposetl classification.
Clinical Aspects and Differential
Diagnosis
Clinically, dcrmoids of the floor of the mouth are frequently quite striking in their appearance. Those presenting themselves intraorally, or suhlingually, may actually displace the tonguc upward to the palate until difficulty in eating, speaking, and even breathing may occur; those presenting extraorally, or submentally, usually appea,r as a pendulous mass beneath the feel, but may feel cystic, mandible. The cysts generally have :I “doughlike” depending on consistency of the contents, which ma.y vary from a cheesy, Hair, nails, keratin, sebaceous-like substance to n more liquefietl material. etc., may be present,28 depending on the type of cyst being dealt wit,h. Some have frank pus present due t,o arute infection. These lesions vary in weight from one gram to several hundrccl grams, a,nd may va.ry in size frorn a small pea-sized growth to one the size of a large grapefruit. Sinus tracts may develop from these cysts to open either intraorally into the floor of the mouth or extraorally into t,he skin beneath the chin. Dermoids may undergo malignant degeneration and may metastasize to lymph nodes.‘, Ii, 20,26 The differential diagnoses of tlermoids of the floor of the mouth include: (I) ranula, (2) unilateral or bilateral blockage of Wharton’s ducts, (3) thyroglossal duct cyst. (4) cystic hygroma,, (5) branchial cleft cysts, (6) acute infection or cellulitis of the floor of the mouth, (7) infections of submaxillary and sublingual glands, (8) benign and malignant tumors of the floor of the mouth and adjacent salivary glands. and (9) normal fat mass in the submental area. The calinical diagnosis is inconclusive.
Treatment The treatment of dermoid cysts of the floor of the mouth is surgical. Aspiration may be used for temporary relief. Aspiration and sclerosing agents, as definitive therapy, are not good; these may lead to even more difficulties, including
DERMOID
1153
CYSTS
acute infection and ccllulitis of the surrounding area. There have been no reports of the use of radiation, but it can readily be assumed that this would not be effective. The surgical approach can he made either intra- or extraorally, depending on the position of the cyst in relation to t,he mylohyoid and geniohyoid muscles. Those lying between geniohyoid and oral mucous membrane (sublingual position) are best removed by an intraoral approach ; those lying between geniohyoid and mylohyoid (submental position) are approached through a transverse incision t,hrough skin, fascia, platysma, and mylohyoid. Usually the cysts are readily shelled out by blunt dissection, with difficulty being encountered only if there has been mattin g down due to previous treatment by aspiration or sclerosing solutions or episotles of infection. Case Reports white married man, Case l.*-Feb. 4, 1954. The patient, S. K., was a 33-year-old which first appeared three years whose chief complaint was “swelling of the throat” earlier. At that time his local physician incised ant1 drained a tender “golf ball-sized” mass intraorally, and a large quantity of whitish, nonfoul material exudetl. The mass did
Fig.
‘.-Case
1.
Preoperative
v&v
showing
submental
mass.
not recur, but the patient went to a clinic for further advice; nothing was found on examination. About ten months later, the mass gradually recurred and increased in size to 8 by 3 cm. Upon bimanual palpation, a doughlike mass could be felt. The mass did not readily displace itself intraorally and appeared to be somewhat more accessible through a skin approach. The family history and laboratory studies were normal. Submandibular dermoid. Preoperative diagnosis: Surgeon : Assistant
Dr. Fred A. Post, Springfield, surgeon:
Date of surgery: *Courtesy
Dr. Irving
Massachusetts.
Meyer.
Feb. 6, 1954.
of Dr. 8’. A. Post, Lesson
Memorial
Hospital,
Springfield,
Massachusetts.
1154
IRVING
MEYER
Fig. 4. Fig. I.-Case 1. Photomicrograph (IOMV-power) of cyst wall showing epithelium. piling up of keratin into the lumen, and chronic inflammation. Fig. 4.-Case 1. Photomicrograph of cyst ~~11 (high-porei”).
stratii%
srluamous
cyst was easily freed by blunt finger dissection and extirpated in toto. The geniohgoid, mylohyoid, and platysma were sutured and a rubber dam drain m-as placed to the depths of the cavity. The skin was then closet1 with black silk. (In retrospect, both surgeons felt that an intraoral approach would have been better than the extraoral approach used.) The patient hat1 an uneventful WCO~~ZI’~ RII(~ has been examined periodically with no evidence of recurrence of the cys:t. Pathology
rc,port:
Gross description: ‘l’hc spwiruen consisted of an oval mass of tissue measuring 5 by 3 by 3 em., the surface of which was generally smooth with small collections of shaggy fibrous tissue present at various points of dissection. The
specimen was generally yellowish pink in color, soft in consistency, and cystic in nature. The mass weighed 29 grams and, upon sectioning, was filled with a cheeselike material. Microscopic aPs~i’iptio~a: Sections consistetl of stratified squamous-cell epithelium with considerable piling of keratin into the lumen. The untlerlying connective tissue was dense and containetl l~loocl vessels, fibrous tissue, and inflammatory cells. Cholesterol slits were present in the connective tissue. Epidermoitl cyst of the floor of the mouth; no evi,I~~CVOSCO~~C aiapt0sis : dence of malignancy. Case 2.*-On Sept. 2, 1951, A. (1., a &Z-year-old unmarried white woman, presented a 10 cm. pendulous mass hanging beneath the mandible and apparently adherent bilaterally The skin was freely movable over the to inner surfaces of the angles of the mandible. mass, which could not be presented intraorally upon submental digital pressure. There was a history of swelling in this area having been incised and tlrained when the patient until the age of 15, when the preswas 2 years oltl. The patient remained free of complaint it, gradually increased to its present size. There was ent mass became very evident;
Fig. 5.-Case ueither patient weight.
2.
Lateral
view
showing
submental
mass
The pain nor discomfort, but the patient was quite conscious of its unsightliness. had been placed on a low-fat, high-protein diet by her physician in order to lose The family history and laboratory findings were normal.
Preoperative suryeon: Assistant
dictptosis:
Dr. Arthur su,rgeon :
Date of swgmy:
Dermoid
cyst of the floor of the mouth.
E. Qramse, Hpriugficld, Dr. Irviug
Massachusetts.
Xicycr.
Sept. 7, 1951.
gcueral anesthesia, with nasal intubation, a high Pindings and procedure : Vndrr transverse incision was matle in the upper neck through skin, subcutaneous tissues, and platysma, ant1 was extencletl back to the angles of the mandible on each side. The mplohyoid muscles were split transversely following the outline11 iucisiou. The anterior belly By blunt dissection, the of each digastrie muscle was exposetl and retracted laterally. present top of the cystic mass was exposed; there was evidence of previous inflammation *Lesson
Memorial
Hospital,
SpringfIeld,
Massachusetts.
1156
1RVING MEYER
1)~ the marked adherence of the mylohyoid muscles to the inferior aspect of the cyst, ant1 geniohyoid muscles to the superior aspect of the cyst. The carotid sheaths of both sides were carefully freed up from the field and retracted. The cyst was then completely freed up from its firm adhesions to the angles of the mandible, base of the tongue, and hyoid bone; it was firmly adherent to and enveloped at the trachea somewhat on each side. The cyst was finally deliverecl intact through the incision without compromising any of the larger nerves or vessels of the neck. The geniohyoitl and mylohyoid muscles, the deep cervical fascia, platysma, and subcutaneous tissues were approximated with catgut suture. The wound was drainetl with a strip of rubber dam, ant1 the skin was closed 1)~ a plastic technique with black silk suture.
Fig.
G.-Case
2. -. Photonlicrograph uell lining, keratin,
(low-power) am1 a hair
The patient had an uneventful postoperative tions have shown no recurrence of the lesion. Pathology
showing cyst wall with stratifled follicle cut in cross section. course and repeatetl
follow-up
squamous examina-
report:
Gross description: The specimen consisted of a large cyst measuring 10 by 5 by 5 em. and containing a granular cheesy material. The cyst wall was tra.nslucent and measured 0.5 mm. in thickness. It presented a smooth inner surface and a roughened outer surface. The specimen weighed 250 grams. AratiHioroscopia aesO~iptiOl~: Hec:t,ions eonwistrd of a st,rip of krratiniertl fied squamous epithelium bortlrred by dense connective tissue containing an occasional chronic: inflammatory ~11. In one area, sevt~ral hair follices were cut in transverse section . L L .,, Microscopic diagnosis: Cyst wall with skin appendages, cal diagnosis of dermoid cyst; no evidence of malignancy. Case 3.*-Aug. 14, 1953. The patient, whose chief complaint was a swelling in the speech. Inasmuch as he was a salesman, this had been given a diet by an internist to help
consistent
with rlini-
H. R., was a 30.year-old married white man floor of the mouth which interfered with his had become a serious problem and the patient him lose weight in this area.
Examination demonstrated a massive swelling in the floor of the mouth which had so raised the body of the tongue palatally that only about 2 cm. of the tip of the tongue The mass could be displaced upward into the mouth by extraoral digital was evident. *Wesson
Memorial
Hospital,
Springfleld,
Massachusetts.
DERMOID
CYSTS
1157
pressure in the submental area. The carunculae of Wharton’s determined. General physical examination and laboratory studies history was noncontributory. Preoperative Surgeon : Assistant
diagnosis: Dr. Irving surgeon
Dermoid
cyst of the floor of the mouth.
Meyer. Dr. Lawrence
:
ducts could scarcely be were normal; the family
Van Selden, Springfield,
Massachusetts.
28, 1953. a traction Findings oxd procedure : Under general anesthesia, with nasal intubation, suture was placed through the body of the anterior third of the tongue. A midline vertical incision was started at the tip of the tongue on the veutrum and carried downward through Date
of surgery:
Aug.
Fig.
Fig.
S.-Case
3.
‘I.--Case
Intraoral
3.
Photograph
photograph
showing
showing
mass
submental
displacing
mass.
the
tongue
to the
palate.
the mucous membrane over;ying the mass for a distance of .I cm. The incision was then extended between the carunculae of Wharton’s ducts to the lingual plate of the alveolus of the mandible. Using blunt dissection, the oral mucous membrane was freed up bilaterally to expose a shiny, silver-white mass beneath. The tlucts of Wharton were identified and retracted from the fielll. The dissection was carried deep around the entire mass, which was approximately the size of a grapefruit. After considerable blunt finger and hemostat dissection, the mass was completely freed from the surrounding musculature and fascia, and was found to be attached b,v a pedicle to the genial tuhercles of the lingual
Vi%. II). Fig. 9.- -(:asc ! 3. weigl zing 325 3m. Fig. 1 o.- -Ck 3e 3. chole stem A-lik e Ins lterial.
Fig.
Il.--Case
3.
Photograph Surgical
Photomicrograyh epitheliunr,
of
surgical
specimen
cut
specimen
measuring
open
to show
(medium-power) hair follicle, an,i
showing sebaceous
contents
a lining g-lnn~ls.
8 by
6
consie iti
Of stratified
5
.‘“I.
of a cht
S~lUarnoUS
DERMOID
CYSTS
1159
aspect of the symphysis of the mandible. The petlicle was clamped between two clamps, cut, and tied. Because of its great size, the mass was tleiivered with some difficulty from the mouth without rupture. The geniohyoitl muscles lying above much stretched mylohyoid muscles were reatlily identified at the base of the large residual spare. The lingual nerves and vessels, as well as genioglossal musculature, were also i(lentifiet1. The lateral walls of the space were brought together with catgut and the oral mucoos membrane was closed with black silk suture. The patient
(lid well postoperatively
and has hall no recurrence
of the lesion.
Gross descripticm.: The specimen consisted of a large cystic structure measuring 8 by 6 by 5 cm. On cut section, the lumen was filled with a cheesy white material; the inner lining was smooth and glistening throughout. The mass weighed 325 grams. It was covered with fine blood vessels on its outer surface. xi~~~.~~pi~ ae.kpti07~ : Sections showed an elongated strip of tissue consisting of dense fibrous connective tissue covered with stratified squamous epiThe epithelium was composed of the different layers from stratum thelium. germinativum to stratum eorneum. There was a notable hpperkeratosis but Hair follicles, some plugged with keratin, were the stratum spinosum was thin. in evidence; sebaceous glands were present in the connective tissue. clinical
NLlicroscopio diagnosis: diagnosis of dermoid
Cyst wall with skin appendages, consistent with There was no evidence of malignancy. cyst.
the
Case 4.x:--April 12 7 1950 . The patient, F. I:., vvab a 2-day-old male Negro infant who, according to the obstetrical service, was born with “two tongues.” The inferior “tongue” which was, on examination, a sublingual, doughlike mass measuring L’ cm. in circumference, The mass was aspirated and 2 C.C. of milky was causing some respiratory embarrassment. The patient was taken home by t,he parents against advice. white fluill was obtained.
Fig.
12.-Case
4.
Clinical
photograph
showing
mass
displacing
tongue
The mass recurred and was again aspiratetl in March, 1950. Bacteriologic aspirations were essentially negative. In April, 1950, the mass recurred brought the infant in for surgical removal of the growth.
upward.
studies of both and the mother
Clinical examination demonstrated a midline saelling beneath the tongue at the base of the lingual frenum. It measured about 2 by 1 cm. on its oral surface and caused a marked upward displacement of thy! tongue. General physical examination and laboratory studies were normal; the family history was noncontributory. Preoperative
Surgeon : Fred
diagnosis: Dr. Irving
*Department of Lucchesi. Chief.
Dermoid
cyst in sublingual
space.
keyever.
Dentistry,
Philadelphia
General
Hospital,
Philadelphia,
Pennsylvania,
1160
IRVING .dSSistant
1)~. Thomas
.Wr{JeO’rls:
Bate of surgery:
April
Meloy
MEYER and Milton
Ivkcr,
Philadelphia,
Pennsylvania.
14, 1950.
Findings and pnxcdwre: Gntler general anesthesia, with nasal intubation, a retraction suture was placed t,hrough the anterior portion of the tongue. B midline incision was started on the ventrurrl of tongue I cm. from the top and carried downward over the protrusion of the cyst between the bilateral carunculae of Wharton’s ducts. The incision was
Fig. 13.-case
4.
Surgical
specimen
measuring
3 by 2 by 2 cm.
Fig. Id.-Case 4. A very low-power view of the cyst wail showing muscle bundles in longi and cross section, lining of the lumen with epithelium, and a connective tissue Capsule surrounding the entire cyst. spread with blunt dissection and carried downward until the body of the clearly exposed. There was a distinct fascial separation between the The geniohyoid and rounding musculature and it was delivered intact. The space and oral at the base of the remaining space were identified. were closed with catgut sutures. The patient made an uneventful recovery and t,here has been no lesion.
cystlike mass was mass and the surmylohyoid muscles mucous membrane recurrence
of the
DERMOID
Pathology
CYSTS
1161
report:
The specimen consisted of a mass measuring 2 by 1 by Gross description: 1 em., encapsulated by a brown fibrous tissue and weighing 3 grams. Upon sectioning the specimen, the cut surface was white and glistenin,g. There was a central lumen, and many small white appendages werr present very slightly raised from the surface. The content of the lumen was a cheesy white material. Fig.
Fig.
15.
16.
Fig. 15.--Case 4. Photomicrograph (medium-power) showing stratified sguamous epitheli urn, hair follicles, sebaceous glands, and keratin in the lumen. l?ig. l&-Case 4. Photomicrograph (medium-power) showing the cyst wall wit .h a *.?A?”ldostratifled columnar epithelial lining, sudoriferous glands, an arteriole, fat tissue, and flbrc bus connective tissue.
1162
IRVING
MEYER
Microscopic desoiptiou : Swt~ions of thv sprc~inlvn ~howetl ali elougat,ed, large lumen lined with a ~callupeci tmlder of it stratifietl squamous epithelium. The epithelium varied in thicknes* from ten to three or four cells, and W:H seen to vary in regions-areas of keratin forulation. pronounced granulosum veils. and cuboidal basal cells which :IW contrastetl tjy lack of keratinization.
Fig.
18.
(me~lium-pobver) showing lining of stratifled e Fig. l’i.-Case 4. Photomicrograph sebaceous glands, and keratin shedding us epithelium, hair follicle in cross section, 1unnen. Fig. 18.-&w 4. Photomicrograph (high-pov’er) of cyst wall (lemonstrating the 11 rolunmar epithelium (nasal type), skeletal muscle. and vas( of ciliated pseudostratified chr mnels.
Portions of the lining Svere comp,o.serl of l~se~~~lo~t~tltifie~l~ colunlnal~, ciliate11 epithelium typical of respiratory (nasal) epitheliun~. The epithelium was not clearly (lelineatetl, iu its entirety, from the uuderThe connective tissue varied lying connective tissue b)- it basement membrane. and contained areas of smooth muscle fibers. in its cellularity and vascularit?;
DERMOID
CYSTS
1163
Comment The four CRWSof cysts of the floor of the mouth presented lend themselves to the classifica.tion suggested in t,his article : Dysontogenetic Cysts of the Floor of the Mouth (a) Epidermoid-Case 1. (b) Dermoid-Cases 2 and 3. (c) Teratoid-Case -I. Case 1 is that of a simple stratified squamous epithelial-lined cyst WIrounded by a connective tissue capsule; this is the epidermoid cyst wit,h its lumen filled with a caseous material composed in part of cholesterol. Cases 2 and 3 are those of stratified squamous epithelial-lined cysts with skin appendages, including hair follicles, sebaceous glands, and sweat glands present in the connective tissue capsule. These are the classic dermoid cysts of the floor of the mouth. Case 4 is that of a teratoid cyst because of the complexity of its conelements are present : (1) epithelial tissues stituent tissues. The following consisting of stratified squamous epithelium and skin appendages, including hair follicles and sebaceous and sudoriferous glands; (2) connective tissues, including the usual vascular and fibrous elements present, striated muscle in both thick bundles and isolated fibers, and large distinct areas of fat tissue; and (3) pseudostratified, columnar, ciliated epithelium of respiratory origin. Summary Dermoid cysts of the floor of the mouth are discussed as to their embryolA classification of these cysts, based ogy, development, and classification. on histopathologic findings, is presented in this article and it is suggested that the word dermoirl be retained as a clinical term for all these developmental cysts of the floor of the mouth. Four cases of dermoid cysts of the floor of the mouth are presented; these were successfully managed surgically without recurrence. A preoperative eva,luation of the location of the cyst in relation to the mylohyoid and geniohyoid muscles, that is, sublingual or submental position, is important in determining whether t.o approach the cyst through an intraoral or an extraoral incision, The clinical diagnosis is inconclusive and the final diagnosis must bc proved by histologic examination.
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MEYER
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