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Macroscopic satellite cyst formation in the odontogenic keratocyst
Macroscopic satellite cyst formation in the odontogenic keratocyst Report
of two
cases
D. A. Keith, B.D.S., P.D.X.R.C.S. KINGS
COLLEGE
HOSPITAL
DENTAL
(Eng.),
London, England
SCHOOL
In view of the current interest in the odontogenic keratocyst, two cases of aggressive cysts in young female patients are described. In both there was macroscopic evidence of satellite cyst formation which illustrates one way in which such cysts may recur after removal. The clinical and histopathologic features of this lesion are briefly reviewed.
T
he clinical features of the odontogenic keratocyst are now well recognized. It may occur at any age, with a peak incidence in the second and third decades, and cases have been reported as early as the first decade of life.2-4 There appears to be a higher incidence in the male. The majority of cysts occur in the mandible, the most common site being the lower third molar area and ascending ramus of the mandible. Expansion is a late finding and occurs more frequently on the buccal aspect; however, lingual plate perforation is frequently noticed. It has been observed that the growth potential of these cysts appears to be in an anteroposterier rather than a buccolingual direction. The presence of a cyst is brought to the attention of the patient usually by infection or looseness of adjacent teeth. The radiologic appearance may be unilocular or multilocular, and resorption of the roots of adjacent teeth is occasionally seen. Aspirate from the cyst cavity either is a thin straw-colored fluid or may have a thick pus-like consistency. It shows a characteristic electrophorectic pattern with a lack of all soluble proteins.6 Bramley and Browne’ and Browne4 have described the histopathologic features. The lining is a continuous layer of regular, stratified squamous epithelium, six to eight cells thick. It is thin and friable, with occasional thicker areas which are associated with irregular epithelial proliferations and possibly with satellite cysts. This latter term is preferred to the term “daughter cyst,” because there is no evidence that satellite cysts arise from the proliferation of the lining of the main cyst. Soskolne and Shear (1967) are of the opinion that satellite cysts arise from adjacent remnants of the dental lamina. Browne4 records satellite cysts in
21
22
Oral Surg. January, 1973
Keith
.
.
.
A.
Fig. 1. Case I. The orthopantomogram shows a well-defined area of bone loss in the of the roots of the second right mandibular premolar and molar region, with resorption premolar and first and second molars.
the capsule in 30 per cent of his cases. The basement membrane is smooth and the basal layer has a well-differentiated and prominent columnar cell layer. The squamous cells show intracellular edema, and there is often an abrupt transition between them and the surface layer of parakeratosis. The fibrous tissue capsule is thin, with few inflammatory cells, except for focal accumulations of plasma cells and lymphocytes. Cholesterol clefts and breaches in the lining epithelium are rare. The significant feature of this type of cyst is its ability to recur, and this property has been the subject of attention and interest for some gears. On the basis of an analysis of 301 odontogenic cysts of all types, of which 5.6 per cent recurred, Fickling5 enumerated the possible causes of recurrence : (1) ineffective original enucleation; (2) failure to ensure healing from the depth of the wound; (3) true budding or daughter cyst formation; and (4) mutliple center potential. Bramley and Brownel postulated that recurrence is a property of the odontogenie cyst itself rather than a failure of technique. Six cysts recurred in their series of 200, and these showed the features of the keratocyst. They listed the possible reasons as : the lining is thin and friable; the lining as adherent; there are bony perforations with the lining lying on soft tissue; daughter cysts may be present. The work of TollerG supports the view that epithelial remnants and satellite cysts, if not enucleated, have the ability to proliferate, which may lead to recurrence. He gives a recurrent rate of 58 per cent. Browne4 reviewed 130 primary and nine recurrent odontogenic keratocysts. There was at least 6 months of follow-up information on 120 of the patients, and in 24.2 per cent the cysts had recurred. The mean rate of recurrence of odontogenic keratocysts is approximately 40 per cent.2, 3
Volume Number
35 1
Satellite
cyst
formation
in odontogenic
Fig. 8. Case I. The per&pica1 radiograph shows the “soap bubble” interradicular bone between the first and second right mandibular premolars.
keratocyst
appearance
23
in the
CASE REPORTS Case
1
Mrs. M. B., a 26-year-old telephone operator, complained of a symptomless lump on the right side of her lower jaw which had been present for about a year. She was a fit young woman with no relevant medical history. There were no skeletal or skin lesions, and no other abnormalities could be detected. Her dental history revealed that a “cyst” had been removed from her right mandible by the school dental surgeon when she was 9 or 10 years old. External examination revealed a painless, bony, hard and smooth swelling on the buccal aspect There of the right mandible in the molar region. It measured 1 by 3 cm. approximately. was no cervical lymphadenopathy and no alteration of labial sensation. Intraorally the swelling could be palpated deep to the buccal sulcus and there was slight lingual expansion. The right mandibular first and second molars were slightly mobile and these teeth, as well as the second premolar, gave a vital response to the electric pulp tester. Radiographs (Fig. 1) showed a well-defined area of bone loss in the right mandible with resorption of the roots of the involved teeth. The area extended down to the lower border of the mandible, but the cortical bone of the lower border was intact. Numerous small areas of bone loss were present in the premolar region giving a %oap bubble” appearance (Fig. 2). The third molar was present and distoangularly impacted. No other lesions were detected in the jaws. A provisional diagnosis of odontogenic keratocyst or possibly ameloblastoma was made. The patient was hospitalized, and, under endotracheal anesthesia, a mucoperiosteal flap was raised from the cuspid to the third molar area. The premolars and molars were extracted and buccal alveolar bone was removed in order to visualize the extent of the lesion. The lining was thin and easily torn, with occasional thickened areas, and the contents were noted to be a straw-colored liquid. The lining was carefully enucleated. A perforation of the lingual bone was observed, and in the interradieular bone in the premolar region numerous soft tissue nodules were present. They could be seen communicating with the main lesion via intrabony channels. This finding necessitated the wide removal of bone in the premolar region. It was noted that the third molar was separated from the lesion by an intact socket wall. The mucosal edges were approximated with silk sutures and a corrugated rubber drain was inserted in the anterior limb of the incision.
24
Keith
Oral Surg. January, 1973
Fig. 8’. Case 1. The epithelial lining has been stripped away from the basement membrane and supporting tissue. There are well-marked columnar basal cells beneath a thin layer of edematous squamous cells. The superficial cells are parakeratinized.
Fdg. 4. Case 2. The lower left molar region.
orthopantomogram
shows a well-defined
area of
bone loss in the
Penicillin was administered and the drain was removed 2 days postoperatively. The patient made a good postoperative recovery with no labial paresthesia, and after 1 week the sutures were removed ; subsequently, the area healed uneventfully. The patient is to be followed as an outpatient, with six monthly radiographic examinations to check for a recurrence. Histopathology. The sections showed a thin and folded cyst wall (Pig. 3). The epithelial lining was three to five cell layers thick and devoid of rete pegs and was torn away in parts from its underlying supporting tissue. The surface was irregularly parakeratinized in parts, with the squamous cells showing marked intracellular edema; and the basal cells, which were mostly columnar, stained deeply with hematoxylin. Immediately beneath the basal cell layer there appeared to be a delicate, relatively acellular collagenous tissue arranged parallel to
Volume 35 Number 1
Satellite
Fig. 5. Case II. The periapical the bone around the molar apices.
cyst formation
radiograph
in odontogenic
shows a similar
“soap
keratocyst
bubble”
appearance
25
in
the surface. This merged on its deeper aspect with more cellular and whorled collagen fibers. Occasionally, foci of mixed inflammatory cells were present and satellite cyst formation was seen. The features were those of an odontogenic keratocyst. Case 2 complained of pain at the left angle of the Mrs. C. B., 30syear-old Negro midwife, mandible. A similar bout of pain 4 years previously had been resolved with antibiotics. The previous medical history was not relevant. On examination she was found to have a tender fluctuant swelling in the buccal sulcus in the molar region which was about 4 cm. long. The second and third molars were abnormally mobile but gave a vital response to the electric pulp tester. There was some slight facial edema and left submandibular lymphadenopathy but no paresthesia. Radiographs (Fig. 4) showed an area of bone loss, extending from the second molar region. The first molar had previously been extracted and the second and third molars had drifted forward. The lower border of the mandible was not affected. Numerous small loculi were present in the interradicular bone between the molars (Fig. 5). An attempt was made to aspirate the contents of the lesion, but the fluid collected was contaminated with blood. A provisional diagnosis of odontogenic keratocyst was made. The patient was hospitalized, and, under endotracheal general anesthesia, a buccal mucopcriosteal flap was raised from the second premolar to the third molar. The cyst lining was exposed and enucleated by dissecting it ofI the inferior dental nerve. The molars were extracted and numerous satellite cysts were noted in the interradicular bone; the involved areas of bone were excised. The buccal plate perforat,ion was found to measure 5 by 3 cm., and the lingual plate perforation was 3 by 1.5 cm. The wound was closed with black silk sutures. The patient made a good postoperative recovery, and the sutures were removed after 1 week. The distal end of the wound broke down but subsequently healed by secondary intenThe area will be radiographed at B-month tion. There was no postoperative paresthesia. intervals in order to check for recurrence. Histopathology. Sections showed a thin-walled cyst (Figure 6) with several satelite cysts (Fig. 7). The very thin stratified squamous epithelial lining was parakeratinized, and in several parts was separated from the basal layer. Some of the basal cells had a loss of polarity of their nuclei and were columnar in appearance. The features were those of an odontogenic keratocyst.
26
Keith
Oral Surg. January, 1973
Fig. 6. Case II. The epithelial surface is parakeratinized and the columnar basal cells show reversed polarity. There is a smooth and thickened basement membrane. (Hematoxylin and eosin, stain. Magnification, x450.) Fig. 7. Case II. Two satellite cysts, showing similar histologic features as the main cyst, which were found partially enclosed by cortical bone. (Hematoxylin and eosin stain. Magnification, x260.)
DISCUSSION
Until the keratocyst was recognized as an entity in itself, such lesions were treated in the same way as other odontogenic cysts, by simple enucleation or, if indicated, marsupialization. Now that the characteristics of keratocysts have been recognized, it is possible to diagnose the true nature of such lesions preoperatively and thus plan the appropriate operative procedure. Furthermore, diligent follow-up examinations to check for recurrences are now obviously necessary. In Case I it may be assumed that the keratocyst recurred after an interval of 16 to 17 years. It is interesting to speculate whether it arose from a deciduous
Volume Number
35 1
Satellite
cyst formation
in odontogenic
keratocyst
27
tooth germ or was associated with a nonvital first or second molar. In both cases, radiographs showed loculation in the adjacent bone, and, at operation, macroscopic satellite cysts were found at these sites. Again in both cases the thin cyst lining had to be dissected off the lingual periosteum where the bone had been perforated. These two cases clearly demonstrate the means by which keratocysts bone which may “recur.” A radical approach should be taken to surrounding may harbor satellite cysts, and adjacent teeth and supporting alveolar bone may have to be removed to prevent such recurrences. SUMMARY
Two cases of odontogenic keratocysts in young patients are described. Both demonstrated macroscopic satellite cyst formation in, addition to locally aggressive characteristics. In view of the clinical appearances, recurrences may be expected. The recent literature is briefly reviewed. I wish to thank Mr. M. Harris, Consultant Dental Surgeon, Kings College Hospital Dental School, for his advice and permission to publish details on patients under his care, and Mr. G. F. Tinsley, Lecturer, Kings College Hospital Dental School, for his advice on the histopathology and for preparing the photomicrographs. REFERENCES
1. Bramley, P. A., and Browne, R. M.: Recurring Odontogenic Cysts, Br. J. Oral Surg. 5: 106-116, 1967. 2. Browne, R. M. : The Odontogenic Keratocyst, Br. Dent. J. 128: 225-231, 1979. 3. Browne, R. M.: The Pathogenesis of the Odontogenic Keratooyst, Proceedings of the Fourth International Conference--International Academy of Oral Pathology, Johannesburg, 1969. Published 1970. 4. Browne, R. M.: The Odontogenic Keratocyst, Br. Dent. J. 131: 249-259, 1971. 5. Fickling, B. W. : Cysts of the Jaw: A Long-term Survey of Types and Treatment, Proc. R. Sot. Med. 58: 847-854, 1965. 6. Toller, P. A.: Origin and Growth of Cysts of the Jaws, Ann. R. Coll. Surg. Engl. 120: 306-335, 1967. Reprint requests to: Dr. D. A. Keith Kings College Hospital Dental Denmark Hill London S.E.5, England
School
of two
cases
D. A. Keith, B.D.S., P.D.X.R.C.S. KINGS
COLLEGE
HOSPITAL
DENTAL
(Eng.),
London, England
SCHOOL
In view of the current interest in the odontogenic keratocyst, two cases of aggressive cysts in young female patients are described. In both there was macroscopic evidence of satellite cyst formation which illustrates one way in which such cysts may recur after removal. The clinical and histopathologic features of this lesion are briefly reviewed.
T
he clinical features of the odontogenic keratocyst are now well recognized. It may occur at any age, with a peak incidence in the second and third decades, and cases have been reported as early as the first decade of life.2-4 There appears to be a higher incidence in the male. The majority of cysts occur in the mandible, the most common site being the lower third molar area and ascending ramus of the mandible. Expansion is a late finding and occurs more frequently on the buccal aspect; however, lingual plate perforation is frequently noticed. It has been observed that the growth potential of these cysts appears to be in an anteroposterier rather than a buccolingual direction. The presence of a cyst is brought to the attention of the patient usually by infection or looseness of adjacent teeth. The radiologic appearance may be unilocular or multilocular, and resorption of the roots of adjacent teeth is occasionally seen. Aspirate from the cyst cavity either is a thin straw-colored fluid or may have a thick pus-like consistency. It shows a characteristic electrophorectic pattern with a lack of all soluble proteins.6 Bramley and Browne’ and Browne4 have described the histopathologic features. The lining is a continuous layer of regular, stratified squamous epithelium, six to eight cells thick. It is thin and friable, with occasional thicker areas which are associated with irregular epithelial proliferations and possibly with satellite cysts. This latter term is preferred to the term “daughter cyst,” because there is no evidence that satellite cysts arise from the proliferation of the lining of the main cyst. Soskolne and Shear (1967) are of the opinion that satellite cysts arise from adjacent remnants of the dental lamina. Browne4 records satellite cysts in
21
22
Oral Surg. January, 1973
Keith
.
.
.
A.
Fig. 1. Case I. The orthopantomogram shows a well-defined area of bone loss in the of the roots of the second right mandibular premolar and molar region, with resorption premolar and first and second molars.
the capsule in 30 per cent of his cases. The basement membrane is smooth and the basal layer has a well-differentiated and prominent columnar cell layer. The squamous cells show intracellular edema, and there is often an abrupt transition between them and the surface layer of parakeratosis. The fibrous tissue capsule is thin, with few inflammatory cells, except for focal accumulations of plasma cells and lymphocytes. Cholesterol clefts and breaches in the lining epithelium are rare. The significant feature of this type of cyst is its ability to recur, and this property has been the subject of attention and interest for some gears. On the basis of an analysis of 301 odontogenic cysts of all types, of which 5.6 per cent recurred, Fickling5 enumerated the possible causes of recurrence : (1) ineffective original enucleation; (2) failure to ensure healing from the depth of the wound; (3) true budding or daughter cyst formation; and (4) mutliple center potential. Bramley and Brownel postulated that recurrence is a property of the odontogenie cyst itself rather than a failure of technique. Six cysts recurred in their series of 200, and these showed the features of the keratocyst. They listed the possible reasons as : the lining is thin and friable; the lining as adherent; there are bony perforations with the lining lying on soft tissue; daughter cysts may be present. The work of TollerG supports the view that epithelial remnants and satellite cysts, if not enucleated, have the ability to proliferate, which may lead to recurrence. He gives a recurrent rate of 58 per cent. Browne4 reviewed 130 primary and nine recurrent odontogenic keratocysts. There was at least 6 months of follow-up information on 120 of the patients, and in 24.2 per cent the cysts had recurred. The mean rate of recurrence of odontogenic keratocysts is approximately 40 per cent.2, 3
Volume Number
35 1
Satellite
cyst
formation
in odontogenic
Fig. 8. Case I. The per&pica1 radiograph shows the “soap bubble” interradicular bone between the first and second right mandibular premolars.
keratocyst
appearance
23
in the
CASE REPORTS Case
1
Mrs. M. B., a 26-year-old telephone operator, complained of a symptomless lump on the right side of her lower jaw which had been present for about a year. She was a fit young woman with no relevant medical history. There were no skeletal or skin lesions, and no other abnormalities could be detected. Her dental history revealed that a “cyst” had been removed from her right mandible by the school dental surgeon when she was 9 or 10 years old. External examination revealed a painless, bony, hard and smooth swelling on the buccal aspect There of the right mandible in the molar region. It measured 1 by 3 cm. approximately. was no cervical lymphadenopathy and no alteration of labial sensation. Intraorally the swelling could be palpated deep to the buccal sulcus and there was slight lingual expansion. The right mandibular first and second molars were slightly mobile and these teeth, as well as the second premolar, gave a vital response to the electric pulp tester. Radiographs (Fig. 1) showed a well-defined area of bone loss in the right mandible with resorption of the roots of the involved teeth. The area extended down to the lower border of the mandible, but the cortical bone of the lower border was intact. Numerous small areas of bone loss were present in the premolar region giving a %oap bubble” appearance (Fig. 2). The third molar was present and distoangularly impacted. No other lesions were detected in the jaws. A provisional diagnosis of odontogenic keratocyst or possibly ameloblastoma was made. The patient was hospitalized, and, under endotracheal anesthesia, a mucoperiosteal flap was raised from the cuspid to the third molar area. The premolars and molars were extracted and buccal alveolar bone was removed in order to visualize the extent of the lesion. The lining was thin and easily torn, with occasional thickened areas, and the contents were noted to be a straw-colored liquid. The lining was carefully enucleated. A perforation of the lingual bone was observed, and in the interradieular bone in the premolar region numerous soft tissue nodules were present. They could be seen communicating with the main lesion via intrabony channels. This finding necessitated the wide removal of bone in the premolar region. It was noted that the third molar was separated from the lesion by an intact socket wall. The mucosal edges were approximated with silk sutures and a corrugated rubber drain was inserted in the anterior limb of the incision.
24
Keith
Oral Surg. January, 1973
Fig. 8’. Case 1. The epithelial lining has been stripped away from the basement membrane and supporting tissue. There are well-marked columnar basal cells beneath a thin layer of edematous squamous cells. The superficial cells are parakeratinized.
Fdg. 4. Case 2. The lower left molar region.
orthopantomogram
shows a well-defined
area of
bone loss in the
Penicillin was administered and the drain was removed 2 days postoperatively. The patient made a good postoperative recovery with no labial paresthesia, and after 1 week the sutures were removed ; subsequently, the area healed uneventfully. The patient is to be followed as an outpatient, with six monthly radiographic examinations to check for a recurrence. Histopathology. The sections showed a thin and folded cyst wall (Pig. 3). The epithelial lining was three to five cell layers thick and devoid of rete pegs and was torn away in parts from its underlying supporting tissue. The surface was irregularly parakeratinized in parts, with the squamous cells showing marked intracellular edema; and the basal cells, which were mostly columnar, stained deeply with hematoxylin. Immediately beneath the basal cell layer there appeared to be a delicate, relatively acellular collagenous tissue arranged parallel to
Volume 35 Number 1
Satellite
Fig. 5. Case II. The periapical the bone around the molar apices.
cyst formation
radiograph
in odontogenic
shows a similar
“soap
keratocyst
bubble”
appearance
25
in
the surface. This merged on its deeper aspect with more cellular and whorled collagen fibers. Occasionally, foci of mixed inflammatory cells were present and satellite cyst formation was seen. The features were those of an odontogenic keratocyst. Case 2 complained of pain at the left angle of the Mrs. C. B., 30syear-old Negro midwife, mandible. A similar bout of pain 4 years previously had been resolved with antibiotics. The previous medical history was not relevant. On examination she was found to have a tender fluctuant swelling in the buccal sulcus in the molar region which was about 4 cm. long. The second and third molars were abnormally mobile but gave a vital response to the electric pulp tester. There was some slight facial edema and left submandibular lymphadenopathy but no paresthesia. Radiographs (Fig. 4) showed an area of bone loss, extending from the second molar region. The first molar had previously been extracted and the second and third molars had drifted forward. The lower border of the mandible was not affected. Numerous small loculi were present in the interradicular bone between the molars (Fig. 5). An attempt was made to aspirate the contents of the lesion, but the fluid collected was contaminated with blood. A provisional diagnosis of odontogenic keratocyst was made. The patient was hospitalized, and, under endotracheal general anesthesia, a buccal mucopcriosteal flap was raised from the second premolar to the third molar. The cyst lining was exposed and enucleated by dissecting it ofI the inferior dental nerve. The molars were extracted and numerous satellite cysts were noted in the interradicular bone; the involved areas of bone were excised. The buccal plate perforat,ion was found to measure 5 by 3 cm., and the lingual plate perforation was 3 by 1.5 cm. The wound was closed with black silk sutures. The patient made a good postoperative recovery, and the sutures were removed after 1 week. The distal end of the wound broke down but subsequently healed by secondary intenThe area will be radiographed at B-month tion. There was no postoperative paresthesia. intervals in order to check for recurrence. Histopathology. Sections showed a thin-walled cyst (Figure 6) with several satelite cysts (Fig. 7). The very thin stratified squamous epithelial lining was parakeratinized, and in several parts was separated from the basal layer. Some of the basal cells had a loss of polarity of their nuclei and were columnar in appearance. The features were those of an odontogenic keratocyst.
26
Keith
Oral Surg. January, 1973
Fig. 6. Case II. The epithelial surface is parakeratinized and the columnar basal cells show reversed polarity. There is a smooth and thickened basement membrane. (Hematoxylin and eosin, stain. Magnification, x450.) Fig. 7. Case II. Two satellite cysts, showing similar histologic features as the main cyst, which were found partially enclosed by cortical bone. (Hematoxylin and eosin stain. Magnification, x260.)
DISCUSSION
Until the keratocyst was recognized as an entity in itself, such lesions were treated in the same way as other odontogenic cysts, by simple enucleation or, if indicated, marsupialization. Now that the characteristics of keratocysts have been recognized, it is possible to diagnose the true nature of such lesions preoperatively and thus plan the appropriate operative procedure. Furthermore, diligent follow-up examinations to check for recurrences are now obviously necessary. In Case I it may be assumed that the keratocyst recurred after an interval of 16 to 17 years. It is interesting to speculate whether it arose from a deciduous
Volume Number
35 1
Satellite
cyst formation
in odontogenic
keratocyst
27
tooth germ or was associated with a nonvital first or second molar. In both cases, radiographs showed loculation in the adjacent bone, and, at operation, macroscopic satellite cysts were found at these sites. Again in both cases the thin cyst lining had to be dissected off the lingual periosteum where the bone had been perforated. These two cases clearly demonstrate the means by which keratocysts bone which may “recur.” A radical approach should be taken to surrounding may harbor satellite cysts, and adjacent teeth and supporting alveolar bone may have to be removed to prevent such recurrences. SUMMARY
Two cases of odontogenic keratocysts in young patients are described. Both demonstrated macroscopic satellite cyst formation in, addition to locally aggressive characteristics. In view of the clinical appearances, recurrences may be expected. The recent literature is briefly reviewed. I wish to thank Mr. M. Harris, Consultant Dental Surgeon, Kings College Hospital Dental School, for his advice and permission to publish details on patients under his care, and Mr. G. F. Tinsley, Lecturer, Kings College Hospital Dental School, for his advice on the histopathology and for preparing the photomicrographs. REFERENCES
1. Bramley, P. A., and Browne, R. M.: Recurring Odontogenic Cysts, Br. J. Oral Surg. 5: 106-116, 1967. 2. Browne, R. M. : The Odontogenic Keratocyst, Br. Dent. J. 128: 225-231, 1979. 3. Browne, R. M.: The Pathogenesis of the Odontogenic Keratooyst, Proceedings of the Fourth International Conference--International Academy of Oral Pathology, Johannesburg, 1969. Published 1970. 4. Browne, R. M.: The Odontogenic Keratocyst, Br. Dent. J. 131: 249-259, 1971. 5. Fickling, B. W. : Cysts of the Jaw: A Long-term Survey of Types and Treatment, Proc. R. Sot. Med. 58: 847-854, 1965. 6. Toller, P. A.: Origin and Growth of Cysts of the Jaws, Ann. R. Coll. Surg. Engl. 120: 306-335, 1967. Reprint requests to: Dr. D. A. Keith Kings College Hospital Dental Denmark Hill London S.E.5, England
School