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Multiple dentigerous cysts of the maxilla and mandible: Report of a case
J Oral Max~llofac 45594497,
Surg
1967
Multiple
Dentigerous Cysts of the Maxilla and Mandible: Report of a Case
LONNIE H. NORRIS, DMD, MPH,* PAOLO PICCOLI, MD, DMD,t MARIA B. PAPAGEORGE, DMD*
AND
vacated by some surgeons in the treatment of large cysts. 14,15However, long-term follow-up is necessary after this procedure because there is a potential malignant transformation of the lesion, and the residual cystic lining may require enucleation. In cases where there is not a strong justification for marsupialization, cystectomy is the preferred treatment. Single dentigerous cysts are well documented in the literature. However, multiple cysts in patients without a syndrome is less common. The following report illustrates a rare case of multiple dentigerous cysts in both the maxilla and mandible involving all the unerupted permanent teeth.
The dentigerous cyst arises from degenerative changes in the reduced enamel epithelium with an accumulation of fluid between the crown of the tooth and the lining epithelium.i*2 In a study by Radden and Read of 368 odontogenic cysts, dentigerous cysts occurred in 17.4% of cases and were more prevalent in the mandibular third molar, mandibular first premolar, and the maxillary incisor areas in order of decreasing frequency.3 The dentigerous cyst can vary in size from a few millimeters to a very large lesion involving the entire body and ramus of the mandible. Perforation of the cortical plate and involvement of the overlying soft tissue can occur. Dentigerous cysts occur twice as often in males than in females.3,4 Surgical treatment of cystic lesions is indicated to prevent local extension from compromising adjacent osseous tissue and later impinging on anatomical structures. Although rare, complications that can occur with the dentigerous cyst are local extension compromising adjacent bone and anatomical structures and neoplastic degeneration.5-8 Partsch introduced the techniques of cystostomy or marsupialization9 (Partsch I) and radical cystectomy (Partsch II),lO respectively. Marsupialization is indicated when there is extensive bony destruction, when the cyst is closely related to important anatomical structures, and in medically compromised patients who cannot tolerate radical surgery.“-I3 The advantages of marsupialization are that adjacent structures can be preserved and following marsupialization it is not necessary to fill the residual cavity to promote bone healing as ad-
Report of a Case A seven-year-old white girl was referred to Tufts University School of Dental Medicine for evaluation and treatment of multiple cyst-like areas around the crowns of all her unerupted permanent teeth. These radiolucent areas were noticed on routine dental radiographs by her dentist. The patient complained of swelling in the left maxilla which had been present for several weeks. The past medical history was noncontributory. The child was in good health with the exception of a recent episode of Varicella. Examination revealed a mixed dentition with the first permanent molars fully erupted and the permanent maxillary lateral incisors erupting buccally. The gingiva was erythematous and there were chicken pox lesions in several areas intraorally (Fig. 1). There were similar vesicular lesions on the patient’s face, neck, arms, and upper torso. Intraorally, there was a slight asymptomatic buccal swelling in the left maxillary primary molar region. No other remarkable findings were apparent. The panoramic radiograph revealed relatively large radiolucent cyst-like areas around the crowns of all unerupted permanent teeth, varying in size from 7.5 to 21 mm (Fig. 2). The lesions had displaced all these teeth apitally, particularly the maxillary canines which were in the infraorbital region and the mandibular canines located at the inferior border. There was no evidence of third molar tooth buds. Skull and chest radiographs and routine laboratory tests were within normal limits. On June 26, 1985, the patient was admitted to the New England Medical Center Hospital. Under general anesthesia the cyst-like areas were first aspirated using a 20gauge needle on a 5 ml syringe; a thick yellowish-white
Received from the Department of Oral and Maxillofacial Surgery, School of Dental Medicine, Tufts University, Boston, Massachusetts. * Assistant Professor. t Clinical Instructor, Department of Odontostomatology, University of Milan, Italy; visiting instructor and faculty member, rifts University. $ Instructor. Address correspondence and reprint requests to Dr. Papageorge: rifts Dental School of Medicine, One Kneeland Street, Boston, MA 02111. 0278-2391187 $0.00 + .25
694
NORRIS ET AL.
695
FIGURE 1 (top). Preoperative clinical photograph of the patient showing the mixed dentition. The first permanent molars are fully erupted and the permanent maxillary lateral incisors arc erupting facially. The gingiva is erythematous and edematous, with obliteration of the normal papillae. Varicella lesions are present in both the maxillary and mandibular gingiva (arrows). FIGURE 2 (botrom). Preoperative panoramic radiograph showing the large radiolucent areas around the crowns of all unerupted permanent teeth. These lesions, varying in size from 7.5-21 mm in diameter, have displaced the teeth apically. The maxillary canines are located close to the infraorbital rim and the mandibular canines are near the inferior border of the mandible.
fluid was obtained. An incision was then made over the unerupted mandibular right permanent second molar, extended along the gingival crevices of the mandibular permanent first molar to the primary canine, and a fullthickness buccal flap was raised in the entire quadrant. This exposed the follicular-like tissue over the crown of the unerupted second molar, that was incised and decompressed. A specimen was taken for histologic study. The cyst-like lining was sutured to the surrounding mucosa, thus marsupializing the lesion. The mandibular right primary canine and first and second molars were very mobile and therefore were extracted. An incision was then made into the exposed cystic lining beneath the extracted primary teeth. The first permanent premolar was floating in the lesion without bony support and was extracted to allow access to the cystic lesion apically to it and surrounding the canine. The lesions around the crowns of the permanent mandibular right second premolar and canine were marsupialized in a similar fashion and biopsies were taken from these lesions. A similar procedure was performed in the left mandible, with extraction of the primary canine, first and second molars and first permanent premolar, and biopsy and marsupilization of the existing cyst. The maxillary right and left primary canines and first and second molars
were extracted due to extensive bony destruction by the underlying cysts. The lesions were marsupialized and biopsies were taken. Again, the permanent right and left first premolars were floating without bony support and, therefore, were extracted allowing access to the cystic area apical to them. In addition, in the left -maxilla, the second premolar was extremely loose and had to be extracted. The second molars were located very high in the maxilla and their location made it impossible to suture the cystic wall to the mucosa. Therefore, incisions were made over the lesions, bony windows were opened, biopsies were taken, and both areas were packed with !U’ Nugauze soaked with bacitracin (500 units/g) ointment. All other marsupialized areas were similarly packed with Nugauze. The patient tolerated the procedure well and was discharged from the hospital the following day with a prescription for elixir of acetaminophen with codeine for pain and a 1Cday course of oral erythromycin. The patient was followed regularly and the packs were advanced weekly. They were removed entirely by six weeks and soft tissue healing was complete by eight weeks. Excellent oral hygiene was maintained by the patient during this period. Regular clinical and radiographic evaluation revealed normal healing. Panoramic radiographs were
696
MULTIPLE
DENTIGEROUS
CYSTS
FIGURE 3 (top left). Panoramic radiograph taken three weeks postoperatively showing the position of the canines, premolars, and second molars. FIGURE 4 (top right). Panoramic radiograph taken nine months postoperatively. The canines and premolars have surfaced intraorally and have obtained a better position. Also, the cystic lesions around these teeth are no longer evident. However. cystic lesions around the second molars are still apparent. FIGURE 5 (bottom right). Panoramic radiograph taken one year postoperatively. There is better alignment and position of the permanent teeth. All teeth except the permanent second molars have erupted intraorally. The cystic lesions around these teeth, however, have decreased in size. Bone density in the area of the marsupialized cysts gradually increased over the course of the year.
taken postoperatively (Fig. 3) and at three-month intervals for one year. Over this period the permanent maxillary and mandibular teeth erupted close to a more normal position. As evident in serial panoramic radiographs, there was bony regeneration and a decrease in the size of the cystic lesions around the crowns of the teeth (Figs. 4, 5). The maxillary and mandibular canines, in particular, which were in an extreme apical and malaligned position prior to surgery, eventually uprighted and erupted in a more normal position without orthodontic intervention. The second premolars which originally were without bony support but were retained entirely in soft tissue have gradually stabilized (Fig. 6).
Histopathology All specimens were similar and consisted of strips and fragments of granulation tissue lined on one surface with degenerating squamous epithelium. A dense intlammatory infiltrate consisting of lymphocytes, plasma cells, histiocytes and neutrophils was seen in the granulation tissue (Fig. 7). The diagnoses were dentigerous cysts. Discussion Multiple cysts have been associated with basal cell nevus syndrome and mucopolysaccaridoses such as Maroteaux-Lamy and Hunter’s syn-
dromes.16-l9 These syndromes were ruled out in our patient because she did not present with any other associated pathology or abnormal laboratory blood values. There are clearly indications for cystectomy rather than marsupialization of dentigerous cysts. Enucleation of the cystic lining is the preferred treatment when the cysts are small and no structures will be endangered by the surgery, or when tumor growth is suspected.*O In cystectomy, the entire epithelial lining is removed, whereas in marsupialization the objective is to release the internal cystic pressure that allows for shrinkage of the cyst. In planning treatment for dentigerous cysts, the risk of tumor development in the cyst wall must be weighed against damage to adjacent anatomical structures. Enucleation involves a shorter treatment period and the entire specimen can be studied histologically. In this case the merits of marsupulization were apparent. To perform cystectomies would have meant removal of all of the permanent dentition except the incisors and first molars, and necessitate complex reconstruction procedures to restore functional masticatory occlusion in a seven-year-old child.
NORRIS ET AL.
697
FIGURE 6 (top). One year postoperative clinical photograph showing complete eruption of the maxillary central incisors and mandibular central and lateral incisors. The canines and premolars have surfaced and are in good alignment at this stage. FIGURE 7 (botrom). Photomicrograph of a dentigerous cyst showing the degenerating squamous cell epithelial lining (arrows) and underlying granulation tissue with a dense infiltrate of inflammatory cells. (Hematoxylin and eosin. Original magnification, X 100.)
References 1. Shafer WG, Hine MK, Levy BM: A Textbook of Oral Pa.thology, 4th ed. Philadelphia, WB Saunders, 1983, pp 258-317 2. Shear M: Cysts of the jaws: recent advances. J Oral Path01 14:43, 1985 3. Radden BG, Reade PC: Odontogenic cysts. A review and a clinical pathological study of 368 odontogenic cysts. Aust Dent J 218, 1973 _ 4. Cabrini RL. Barros RE. Albano H: Cvsts of the iaws: a statistical analysis. J Oral Surg 28:485, 1970 ” 5. Stanley HR, Diehl DL: Ameloblastoma potential of follicular cysts. Oral Path01 20:260, 1965 6. Browne RM, Gough N: Malignant change in the epithelium lining of odontogenic cysts. Cancer 29: 1199, 1972 7. Gardner AF: A survey of odontogenic cysts and their relationship to squamous cell carcinoma. J Can Dent Assoc 41:161, 1975 8. Norris L, Baghaei-Rad M, Maloney P, et al: Bilateral maxillary squamous odontogenic tumors and the malignant transformation of a mandibular radiolucent lesion. J Oral Maxillofac Surg 42~827, 1984 9. Partsch K: Uber kieFerzysten. Dtsch Wschr Zahnheilk 7:271, 1895 10. Partsch K: Zur behandlung der kieFerzysten. Dtsch Mischr Zahnheilk 20:252, 1910
11. Yoshikawa Y, Nakajima T, Kaneshiro S, et al: Effective treatment of the postoperative maxillary cysts by marsupialization. J Oral Maxillofac Surg 40:487, 1982 12. Matsumura T, Ishihara Y, Hayashi T, et al: Preservation of the permanent tooth in the follicular cyst by marsupialization: report of two cases. Jpn J Oral Surg 25:251, 1979 13. Jacobi R: Spontaneous repositioning of displaced molars after marsupialization of a dentigerous cyst. J Am Dent Assoc 102:655, 1981 14. Baumann M: Langzeilterfahrungen mit der marsupialization grosser unterkieferzysten zur mundhole, 86: 1280, 1976 15. Olson RE, Thomsen S, Lin L-M: Odontogenic keratocyst treated by the Partsch operation and delayed enucleation: report of case. J Am Dent Assoc 94:321, 1977 16. Gorlin RJ, Vickers RA, Kelln E, et al: The multiple basal cell nevi syndrome. Cancer 18:89, 1965 17. Skaug N, Hofstad T: Demonstration of glycosaminoglycans in fluid from jaw cysts. Acta Path01 Microbial Stand 80:285, 1972 18. Roberts MW, Costantopoulos G, Donahue AH: Occurrence of multiple dentigerous cysts in a patient with the Maroteaux-Lamy syndrome (mucopolysaccharidosis, type VI). Oral Surg 58:169, 1984 19. Lustman J, Bimstein E, Yatziv S: Dentigerous cysts and radiolucent lesions of the jaw associated with Hunter’s syndrome. J Oral Surg 33:679, 1975 20. Russell AY: Conversative management of bone cysts in children and adults. J Am Dent Assoc 23:1719, 1936
Surg
1967
Multiple
Dentigerous Cysts of the Maxilla and Mandible: Report of a Case
LONNIE H. NORRIS, DMD, MPH,* PAOLO PICCOLI, MD, DMD,t MARIA B. PAPAGEORGE, DMD*
AND
vacated by some surgeons in the treatment of large cysts. 14,15However, long-term follow-up is necessary after this procedure because there is a potential malignant transformation of the lesion, and the residual cystic lining may require enucleation. In cases where there is not a strong justification for marsupialization, cystectomy is the preferred treatment. Single dentigerous cysts are well documented in the literature. However, multiple cysts in patients without a syndrome is less common. The following report illustrates a rare case of multiple dentigerous cysts in both the maxilla and mandible involving all the unerupted permanent teeth.
The dentigerous cyst arises from degenerative changes in the reduced enamel epithelium with an accumulation of fluid between the crown of the tooth and the lining epithelium.i*2 In a study by Radden and Read of 368 odontogenic cysts, dentigerous cysts occurred in 17.4% of cases and were more prevalent in the mandibular third molar, mandibular first premolar, and the maxillary incisor areas in order of decreasing frequency.3 The dentigerous cyst can vary in size from a few millimeters to a very large lesion involving the entire body and ramus of the mandible. Perforation of the cortical plate and involvement of the overlying soft tissue can occur. Dentigerous cysts occur twice as often in males than in females.3,4 Surgical treatment of cystic lesions is indicated to prevent local extension from compromising adjacent osseous tissue and later impinging on anatomical structures. Although rare, complications that can occur with the dentigerous cyst are local extension compromising adjacent bone and anatomical structures and neoplastic degeneration.5-8 Partsch introduced the techniques of cystostomy or marsupialization9 (Partsch I) and radical cystectomy (Partsch II),lO respectively. Marsupialization is indicated when there is extensive bony destruction, when the cyst is closely related to important anatomical structures, and in medically compromised patients who cannot tolerate radical surgery.“-I3 The advantages of marsupialization are that adjacent structures can be preserved and following marsupialization it is not necessary to fill the residual cavity to promote bone healing as ad-
Report of a Case A seven-year-old white girl was referred to Tufts University School of Dental Medicine for evaluation and treatment of multiple cyst-like areas around the crowns of all her unerupted permanent teeth. These radiolucent areas were noticed on routine dental radiographs by her dentist. The patient complained of swelling in the left maxilla which had been present for several weeks. The past medical history was noncontributory. The child was in good health with the exception of a recent episode of Varicella. Examination revealed a mixed dentition with the first permanent molars fully erupted and the permanent maxillary lateral incisors erupting buccally. The gingiva was erythematous and there were chicken pox lesions in several areas intraorally (Fig. 1). There were similar vesicular lesions on the patient’s face, neck, arms, and upper torso. Intraorally, there was a slight asymptomatic buccal swelling in the left maxillary primary molar region. No other remarkable findings were apparent. The panoramic radiograph revealed relatively large radiolucent cyst-like areas around the crowns of all unerupted permanent teeth, varying in size from 7.5 to 21 mm (Fig. 2). The lesions had displaced all these teeth apitally, particularly the maxillary canines which were in the infraorbital region and the mandibular canines located at the inferior border. There was no evidence of third molar tooth buds. Skull and chest radiographs and routine laboratory tests were within normal limits. On June 26, 1985, the patient was admitted to the New England Medical Center Hospital. Under general anesthesia the cyst-like areas were first aspirated using a 20gauge needle on a 5 ml syringe; a thick yellowish-white
Received from the Department of Oral and Maxillofacial Surgery, School of Dental Medicine, Tufts University, Boston, Massachusetts. * Assistant Professor. t Clinical Instructor, Department of Odontostomatology, University of Milan, Italy; visiting instructor and faculty member, rifts University. $ Instructor. Address correspondence and reprint requests to Dr. Papageorge: rifts Dental School of Medicine, One Kneeland Street, Boston, MA 02111. 0278-2391187 $0.00 + .25
694
NORRIS ET AL.
695
FIGURE 1 (top). Preoperative clinical photograph of the patient showing the mixed dentition. The first permanent molars are fully erupted and the permanent maxillary lateral incisors arc erupting facially. The gingiva is erythematous and edematous, with obliteration of the normal papillae. Varicella lesions are present in both the maxillary and mandibular gingiva (arrows). FIGURE 2 (botrom). Preoperative panoramic radiograph showing the large radiolucent areas around the crowns of all unerupted permanent teeth. These lesions, varying in size from 7.5-21 mm in diameter, have displaced the teeth apically. The maxillary canines are located close to the infraorbital rim and the mandibular canines are near the inferior border of the mandible.
fluid was obtained. An incision was then made over the unerupted mandibular right permanent second molar, extended along the gingival crevices of the mandibular permanent first molar to the primary canine, and a fullthickness buccal flap was raised in the entire quadrant. This exposed the follicular-like tissue over the crown of the unerupted second molar, that was incised and decompressed. A specimen was taken for histologic study. The cyst-like lining was sutured to the surrounding mucosa, thus marsupializing the lesion. The mandibular right primary canine and first and second molars were very mobile and therefore were extracted. An incision was then made into the exposed cystic lining beneath the extracted primary teeth. The first permanent premolar was floating in the lesion without bony support and was extracted to allow access to the cystic lesion apically to it and surrounding the canine. The lesions around the crowns of the permanent mandibular right second premolar and canine were marsupialized in a similar fashion and biopsies were taken from these lesions. A similar procedure was performed in the left mandible, with extraction of the primary canine, first and second molars and first permanent premolar, and biopsy and marsupilization of the existing cyst. The maxillary right and left primary canines and first and second molars
were extracted due to extensive bony destruction by the underlying cysts. The lesions were marsupialized and biopsies were taken. Again, the permanent right and left first premolars were floating without bony support and, therefore, were extracted allowing access to the cystic area apical to them. In addition, in the left -maxilla, the second premolar was extremely loose and had to be extracted. The second molars were located very high in the maxilla and their location made it impossible to suture the cystic wall to the mucosa. Therefore, incisions were made over the lesions, bony windows were opened, biopsies were taken, and both areas were packed with !U’ Nugauze soaked with bacitracin (500 units/g) ointment. All other marsupialized areas were similarly packed with Nugauze. The patient tolerated the procedure well and was discharged from the hospital the following day with a prescription for elixir of acetaminophen with codeine for pain and a 1Cday course of oral erythromycin. The patient was followed regularly and the packs were advanced weekly. They were removed entirely by six weeks and soft tissue healing was complete by eight weeks. Excellent oral hygiene was maintained by the patient during this period. Regular clinical and radiographic evaluation revealed normal healing. Panoramic radiographs were
696
MULTIPLE
DENTIGEROUS
CYSTS
FIGURE 3 (top left). Panoramic radiograph taken three weeks postoperatively showing the position of the canines, premolars, and second molars. FIGURE 4 (top right). Panoramic radiograph taken nine months postoperatively. The canines and premolars have surfaced intraorally and have obtained a better position. Also, the cystic lesions around these teeth are no longer evident. However. cystic lesions around the second molars are still apparent. FIGURE 5 (bottom right). Panoramic radiograph taken one year postoperatively. There is better alignment and position of the permanent teeth. All teeth except the permanent second molars have erupted intraorally. The cystic lesions around these teeth, however, have decreased in size. Bone density in the area of the marsupialized cysts gradually increased over the course of the year.
taken postoperatively (Fig. 3) and at three-month intervals for one year. Over this period the permanent maxillary and mandibular teeth erupted close to a more normal position. As evident in serial panoramic radiographs, there was bony regeneration and a decrease in the size of the cystic lesions around the crowns of the teeth (Figs. 4, 5). The maxillary and mandibular canines, in particular, which were in an extreme apical and malaligned position prior to surgery, eventually uprighted and erupted in a more normal position without orthodontic intervention. The second premolars which originally were without bony support but were retained entirely in soft tissue have gradually stabilized (Fig. 6).
Histopathology All specimens were similar and consisted of strips and fragments of granulation tissue lined on one surface with degenerating squamous epithelium. A dense intlammatory infiltrate consisting of lymphocytes, plasma cells, histiocytes and neutrophils was seen in the granulation tissue (Fig. 7). The diagnoses were dentigerous cysts. Discussion Multiple cysts have been associated with basal cell nevus syndrome and mucopolysaccaridoses such as Maroteaux-Lamy and Hunter’s syn-
dromes.16-l9 These syndromes were ruled out in our patient because she did not present with any other associated pathology or abnormal laboratory blood values. There are clearly indications for cystectomy rather than marsupialization of dentigerous cysts. Enucleation of the cystic lining is the preferred treatment when the cysts are small and no structures will be endangered by the surgery, or when tumor growth is suspected.*O In cystectomy, the entire epithelial lining is removed, whereas in marsupialization the objective is to release the internal cystic pressure that allows for shrinkage of the cyst. In planning treatment for dentigerous cysts, the risk of tumor development in the cyst wall must be weighed against damage to adjacent anatomical structures. Enucleation involves a shorter treatment period and the entire specimen can be studied histologically. In this case the merits of marsupulization were apparent. To perform cystectomies would have meant removal of all of the permanent dentition except the incisors and first molars, and necessitate complex reconstruction procedures to restore functional masticatory occlusion in a seven-year-old child.
NORRIS ET AL.
697
FIGURE 6 (top). One year postoperative clinical photograph showing complete eruption of the maxillary central incisors and mandibular central and lateral incisors. The canines and premolars have surfaced and are in good alignment at this stage. FIGURE 7 (botrom). Photomicrograph of a dentigerous cyst showing the degenerating squamous cell epithelial lining (arrows) and underlying granulation tissue with a dense infiltrate of inflammatory cells. (Hematoxylin and eosin. Original magnification, X 100.)
References 1. Shafer WG, Hine MK, Levy BM: A Textbook of Oral Pa.thology, 4th ed. Philadelphia, WB Saunders, 1983, pp 258-317 2. Shear M: Cysts of the jaws: recent advances. J Oral Path01 14:43, 1985 3. Radden BG, Reade PC: Odontogenic cysts. A review and a clinical pathological study of 368 odontogenic cysts. Aust Dent J 218, 1973 _ 4. Cabrini RL. Barros RE. Albano H: Cvsts of the iaws: a statistical analysis. J Oral Surg 28:485, 1970 ” 5. Stanley HR, Diehl DL: Ameloblastoma potential of follicular cysts. Oral Path01 20:260, 1965 6. Browne RM, Gough N: Malignant change in the epithelium lining of odontogenic cysts. Cancer 29: 1199, 1972 7. Gardner AF: A survey of odontogenic cysts and their relationship to squamous cell carcinoma. J Can Dent Assoc 41:161, 1975 8. Norris L, Baghaei-Rad M, Maloney P, et al: Bilateral maxillary squamous odontogenic tumors and the malignant transformation of a mandibular radiolucent lesion. J Oral Maxillofac Surg 42~827, 1984 9. Partsch K: Uber kieFerzysten. Dtsch Wschr Zahnheilk 7:271, 1895 10. Partsch K: Zur behandlung der kieFerzysten. Dtsch Mischr Zahnheilk 20:252, 1910
11. Yoshikawa Y, Nakajima T, Kaneshiro S, et al: Effective treatment of the postoperative maxillary cysts by marsupialization. J Oral Maxillofac Surg 40:487, 1982 12. Matsumura T, Ishihara Y, Hayashi T, et al: Preservation of the permanent tooth in the follicular cyst by marsupialization: report of two cases. Jpn J Oral Surg 25:251, 1979 13. Jacobi R: Spontaneous repositioning of displaced molars after marsupialization of a dentigerous cyst. J Am Dent Assoc 102:655, 1981 14. Baumann M: Langzeilterfahrungen mit der marsupialization grosser unterkieferzysten zur mundhole, 86: 1280, 1976 15. Olson RE, Thomsen S, Lin L-M: Odontogenic keratocyst treated by the Partsch operation and delayed enucleation: report of case. J Am Dent Assoc 94:321, 1977 16. Gorlin RJ, Vickers RA, Kelln E, et al: The multiple basal cell nevi syndrome. Cancer 18:89, 1965 17. Skaug N, Hofstad T: Demonstration of glycosaminoglycans in fluid from jaw cysts. Acta Path01 Microbial Stand 80:285, 1972 18. Roberts MW, Costantopoulos G, Donahue AH: Occurrence of multiple dentigerous cysts in a patient with the Maroteaux-Lamy syndrome (mucopolysaccharidosis, type VI). Oral Surg 58:169, 1984 19. Lustman J, Bimstein E, Yatziv S: Dentigerous cysts and radiolucent lesions of the jaw associated with Hunter’s syndrome. J Oral Surg 33:679, 1975 20. Russell AY: Conversative management of bone cysts in children and adults. J Am Dent Assoc 23:1719, 1936