HEREDITY KURT
H.
THOMA,
AND
D.M.D.,
AND
CYST FORMATION FRED
R.
BLCMENTHAI~,
TI.IN1.U.
T
HE heredity of abnormalities of the tooth system has been studied by Keeler (1935),0 who reported the occurrence of brown, transparent, fused, and impacted teeth, besides such jaw deformities as prognathism and hairlip occuring in members of several generations of families. These characteristics are passed to successive generations by genes, units of inheritance which are found in exceedingly small, filamentous structures known as chromosomes in the nuclei of either the male or female cells from which the individual arises. That several members of a family develop cysts has probably been observed more frequently than the literature indicates. The formation of a globulomaxillary cyst in a mother, and later in her daughter has been reported (Thoma, 1944) .t The rarity of this type of cyst should preclude that this occurrence was a coincidence. It has also been noted that some individuals have a constitutional disposition to cyst formation, several cysts frequently being found in one person in different parts of the upper and lower jaws. Less common is the formation of cysts in succession. One of us (Thoma) had a patient with a series of cysts which arose one after another. He was first seen in 1938 when he had a. multilocular cyst in the mandible which extended from the left second incisor to the first premolar; in 1940 he had a follicular cyst in the posterior part of thle right maxilla extending from the right second premolar to the second molar; in 1941 a multilocular cyst was excised from the left maxilla, which extended from the second incisor to the canine ; and when last seen in 1942 a small maxilloglobular cyst was seen developing between the right maxillary second incisor and canine tooth. The x-ray is extremely interesting because it shows the point of origin of this type of fissural cyst (Fig. 545). This paper is a report of the occurrence of cysts in three generations of one Two of the patients family seen for orthodontic care by one of us (Blumenthal). were operated on in our clinic by one of us (Thoma), and some of the others at an outside hospital. One patient was treated by Dr. B. G. Anderson of Yale University, and one by Dr. J. W. Kemper in Ann Arbor, Michigan. It is interesting to note that the cysts in this family tend to be multiple, denoting a very strong hereditary character. The family pedigree is shown in Fig. 546. In the first generation, the grandfather had excellent teeth but developed a cyst in the lower left canine region, which was removed at the age of 78 years by Dr. O’Sullivan of Fall River; the grandmother had very poor teeth. Little else is known about their dental history. In the second generation, there were two sons and three daughters, one son and two daughters had cysts, and the *Keeler, fThoma,
C. E.: K. H.:
Heredity in Dentistry, Oral Pathology, ed.
Dental Cosmos 77: 1147, 1935. 2, St. Louis. 1944, The C. V. Mosby $78
Co.,
p. 873.
274
Thoma, Smith,
BOSCO,Blumenthal,
and Goldman
remaining son and daughter were heterozygotes, the dental characteristic being recessive and transmitted to their children who developed cysts. One son had three children, the dental condition of whom is not known ; the other son had three children of whom one daughter had three cysts. One daughter had two children, one with three cysts ; the other was not investigated. Another daughter had five children, two w&h cysts. The third daughter was operated on by one
Fig.
Fig.
546.-Pedigree
of
545.-Very
family hatched
early
formation
of
maxilloglobular
with cysts. Squares indicate portions, members affected with
cyst.
males; cysts.
circles,
females;
and
of us (Thoma thirteen years ago for an infected dentigerous cyst connected with an unerupted left canine in the upper jaw. She had eleven children, five boys and six girls. We were fortunate to be able to make an investigation of them with the kind cooperation of Dr. J. F. Boyd, roentgenologist in Providence, R. I. One member of the family was not available as she was living in another part of the country, but of the ten children, cysts were found in four, one son and three daughters. It is interesting to note that the mother had a dentigerous cyst
Clinic
of ~~a~ssachusetts
General
275
Hospital
formed from the left maxillary canine, while three of her children, one boy and two girls, had similar cysts, all formed from the same tooth on the other side, the right maxillary canine. Unfortunately we were not able to get x-rays of all members of this family, therefore, only case reports of the patients we treated are included. Case She She ago. She
116
L. G. (469197), a 44year-old woman, complained of a sinus discharging had had all her teeth extracted eight years ago, and has been wearing had had a cyst in the mandible for several years, but it gave no trouble Since then there has been a daily discharge from a sinus communicating had had a similar cyst removed, but otherwise had been in good health.
547), (Fig.
X-ray examination and a multilocular 548). In addition,
showed a multilocular cyst in the ramus dentigerous cyst formed from the third there was a cyst in the left maxilla.
into the mouth. a denture since. until three years with the cyst.
of the left mandible (Fig. molar in the right mandible
On Oct. 30, 1944, under gas, oxygen, and ether anesthesia, the oral cavity for operation in the usual manner. The cyst in the maxilla was operated on first. was made in the left buccoalveolar sulcus, and a mucoperiosteal flap prepared Some of the cystic fluid escaped and the cyst membrane came into view. It was the bone cavity and removed. A thin, bony partition, separating the cyst from sinus, was excised, after which a window was made in the nasoantral wall by antral rasp. A dressing was inserted into the sinus with the end protruding nostril. The incision was closed with sutures. .
was prepared An incision and retracted. detached from the maxillary means of an through the
The cyst in the right ramus was excised next. The incision was made along the anterior border of the ascending ramus, and extended along the alveolar crest. The mucoperiosteum was detached from the bone, and after the very thin bony wall of the cyst was removed the The tooth was loosened with elevators and removed with the cyst sac could be detached. cyst attached to it. Sulfanilamide powder was dusted into the cyst cavity, and a boric strip inserted. It was felt that this operation greatly weakened the jaw, so the other cyst was left to be removed at another time. Examination of the tissue showed a tooth with red-gray tissue attached at the neck. This represented a cyst sac which was lined with squamous epithelium. The postoperative course was uneventful and the patient was discharged on Nov. 3, 1944. She was then seen at regular intervals for dressings until the entire cyst cavity in the mandible She was given a syringe to irriga.te the wound until had been covered with granulation tissue. it was completely healed. Case M. M., submaxillary
a young girl, lymph nodes
117
complained of pain and swelling on the left side of the face. The were enlarged and tender, and had been present for about one year.
X-ray examination showed the tooth was absent (Fig. 549). addition, there were two unerupted
a large cystic On the right third molars
defect on the a dentigerous in the maxilla.
left cyst
in the third molar region; was found (Fig. 550). In
On June 7 the patient was operated on at the Brooks Hospital. Under intravenous pentothal sodium anesthesia, the oral cavity was prepared for operation in the usual manner. An incision was made on the left over the alveolar crest starting from the anterior border of the ascending ramus, and extending to the distal surface of the second molar. From here a vertical extension was made on the outer side of the jaw. It was necessary to remove some of the bone with rongeurs and chisels in order to expose the cyst completely. The sac was dissected away from the cyst cavity and removed. A boric strip was placed into the wound. A a great
similar amount
incision was made on the of thick , yellow material
right, and containing
a cyst sac cholesterin
exposed crystals
and incised. was aspirated,
After the
276
Thoma, Smith, Bosco, Blumenthal,
Fig.
Fig. Fig.9 . 547
and
548.-Case
and Goldman
547.
548.
116. X-rays showing multilocular mandible, and (8) in the right
dentigerous mandible.
cysts,
(1)
in
the
left
Clinic
of
Massachusetts
277
General Hospital
It wai loosened with elevators and removed crown of the unerupted third molar was seen. with the cyst sac attached. The two unerupted third molars were excised next. The one pn the right was found to be surrounded by a cyst sac formed from the follicle; on the left the follicle was of normal size. for
Figs.
The patient was allowed dressings until the wounds
549
and
550.-Case
117.
to go home had healed.
Follicular (8) in
on the
Fig.
549.
Fig.
550.
cyst (1) the right
sixth
postoperative
in the left mandible.
mandible,
day,
and
and
was
dentigerous
followed
cyst
278
Thoma, Smith,
BOSCO,Blumenthal,
und Goldman
Case 118 B.
P. was seen recently, and stated that he had been operated on four times for the removal of cysts. The first was a dentigerous cyst of the upper right maxilla which formed from an unerupted canine (e, Fig. 551). This cyst was typical of those inherited by other members of the family. The second cyst occurred in the lower left third molar region. This also was a dentigerous cyst containing a third molar tooth, The third was a dentigerous cyst in the lower right third molar area. The fourth cyst wa,s discovered when a swelling developed The patient had a soar in this area. on the upper jaw. The location of the cyst and the fact that it did not contain a tooth would lead one to believe that it was a globulomaxillary cyst. The two premolars, canine, and incisors were in normal position in this segment of the jaw. The fifth cyst was in the left maxilla in the region of the third molar tooth, which had never formed. This cyst had been removed, but a few days ago a swelling appeared at the tuberosity followed by a discharge into the mouth, which brought the patient to the office for examination. A fistula was found on the posterior surface of the tuberosity into which a probe could be inserted for about % inch. An x-ray taken with the probe inserted showed the presence of a residual cyst cavity. The patient was given an admission for excision.
Fig.
551.-Case
118.
Dentigerous
cyst
Case L.
P., a young woman, complained region in the upper jaw. Examination be inserted for a considerable distance,
of
maxilla
with
unerupted
canine,
C.
119
of a purulent discharge from the right third showed a fistula discharging some pus. A probe and a hard, smooth object could be felt.
molar could
X-rays taken from Water’s position (Fig. 552) showed an unerupted, partly formed In the lateral view (Fig. 553) the tooth was seen third molar, with a radiopaque antrum. below the floor of the orbit. There was some evidence of a narrow cystic tract. A lateral jaw film (Fig. 554) showed a second cyst of large dimensions in the ramus of the jaw. The operation was performed at the Brooks Hospital, July 17, under avertin and ether anesthesia. An incision was made high in the buccoalveolar sulcus. The mucosa was stripped from the bone behind the zygomatic process of the maxilla, and removed by means of a chisel. The cyst came into view and the tooth could be felt. It was easily detached from
Clinic
Pig.
552.-Case
119.
Fig.
279
of Massachzcsetts General Hospital
Dent&emus
553.-Case
119.
cyst
in
Dentigerous
maxilla
cyst,
in
posteroanterior
lateral
view.
vie
!W.
280
Il’homa, flmith,
its location and removed with strip saturated. with petrolatum. of the for
The mandibular ramus, retracting
The patient postoperative’care.
was
the
BOSCO,Blumenthal, cyst
sac
attached.
The
and Goldman wound
was
packed
with
a gauze
cyst
was excised next through an incision made along the anterior border the soft tissue to each side. The cyst sac could be easily removed. discharged from the hospital after five days, and was seen at the office
Rig.
554.-Case
119.
Follicular Case
cyst
in
ramus.
120
E. P., a young girl, was seen on April 4 for a general x-ray examination .l’he right maxillary canine tooth had never erupted. X-rays revealed a large (Fig. 555) in the upper jaw formed from an unerupted canine. The second premolar had been somewhat displaced.
of her teeth. dentigerous cyst incisor and first
On April 5 at the Brooks Hospital, under gas, oxygen, and ether anesthesia, the oral tissues were prepared in the usual manner. A curved incision was made along the gingival margin of the palate. The mucoperiosteum was detached from the thin bony wall of the cyst, and after the bone was removed with rongeur forceps, the cyst sac was exposed and detached from the cyst cavity. It ruptured, and a clear, yellow fluid escaped. When the cyst sac was opened, this brought into view the crown of the unerupted tooth which could be loosened with elevators. The tooth was removed with the cyst sac intact. The mucoperiosteum was pressed into the bottom of the cavity and held there by means of a boric strip. A wire was drawn over it to hold the strip in place, and the ends were anchored on each side by winding them around a tooth through the interdental space. The patient was discharged from the hospital after four days. Postoperative treatment consisted of changing the dressing and irrigating the wound with saline solution. After six days the entire bone cavity was covered by tissue. The patient was then given a syringe to irrigate the wound after meals. She was seen again two years later when a new cyst had developed in the right mandible. There was no clinical evidence except a displacement of the two adjoining teeth, the second
Clinic
of
Massachusetts
incisor and canine. The x-ray showed an ovoid-shaped cystic second incisor causing a divergence of the roots, and extending the body of the mandible (Fig. 556). Excision was advised.
Fig. Fig. Fig.
555.-Case 556.-Case
120. 120.
555. Dentigerous F’ollicular
281
General Hospital area from
between the canine the alveolar process
Fig.
and into
556.
cyst in maxilla. cyst in -mandible.
On July 3, under gas, oxygen, and ether anesthesia at the Brooks Hospital, an. inverted The mucosa U-shaped incision was made on the outer surface of the mandible over the cyst. was dissected away from the bone, and by means of a chisel, the outer cortex which was quite solid was chiseled away to expose the cyst sac. This was grasped with Alyce forceps, and after it was detached from its bony wall, it was removed. The cyst cavity was examined The mucosa was placed into the bottom and no evidence of tooth involvement could be found. of the cavity, ‘and a boric strip inserted. The patient was discharged from the hospital after three days. The postoperative treatment consisted of changing the pack and irrigating the wound. After one week the patient was given a syringe to keep the wound free of food and debris while it healed. This cyst must have formed from an epithelial cell rest between the two teeth, and therefore should be classified a paradontd cyst.