- Email: [email protected]
Multiple calcifying hyperplastic dental follicles
Multiple calcifying hyperplastic dental follicles David G. Gardner, DDS, MSD, a and Bryan Radden, BDSc, PhD, MDSc, FDSRCS, FRCPath, b Denver, Colo. and Melbourne, Australia UNIVERSITY OF COLORADO SCHOOL OF DENTISTRY AND SCHOOL OF DENTAL SCIENCE, UNIVERSITY OF MELBOURNE
This article describes two additional examples of this unusual condition; two others were reported previously. The microscopic features are those of the hyperplastic dental follicles that occur in regional odontodysplasia. However, the teeth are not defective, as in regional odontodysplasia. This rare condition is sufficiently distinctive to be considered a pathologic entity. (ORAL SURGORALMED ORALPATHOLORALRADIOL[NDOD 1995;79:603-6)
To date, two examples have been published 1, 2 of enlarged dental follicles that were associated with multiple unerupted teeth and that exhibited abundant calcifications and rests of odontogenic epithelium. In both cases, the authors considered them to be hamartomas and likened their histologic appearance to that of the central odontogenic fibroma ( W H O type) 3 and of the hyperplastic follicles of regional odontodysplasia. 4 The purpose of the present article is to report two additional examples and to discuss the nature of this unusual condition. CASE REPORTS Case 1 This patient was a 26-year-old white man whose mandibular left second and third molar teeth had not erupted (Fig. 1). Both teeth exhibited circumcoronal radiolucencies that were approximately 6 mm wide and that were found at surgery to consist of dense fibrous connective tissue. Microscopic findings.The lesions consisted of relatively dense, moderately cellular, fibrous connective tissue, throughout which were scattered numerous rests of odontogenic epithelium and whorled structures composed of fibroblasts, collagen fibers arranged concentrically, and small calcifications similar to those that have been described as Type "A" calcifications in the hyperplastic follicles of regional odontodysplasia (Figs. 2 and 3). In many areas these calcifications had coalesced into larger bodies in which the original small calcifications could be clearly identified. In a few areas the whorled structures exhibited only granular calcifications. Elsewhere, the Type "A" calcifications, which did not polarize, had become incorporated into an eosinophilic material (Fig. 4), the fibrillar nature of which was demonstrable by polarization microscopy (Fig. 5). These Type "B" calcifications (Figs. 4 and 5) also exhibited aprofessor, Divisionof Oral Pathologyand Oncology,Universityof Colorado School of Dentistry. bSenior Associate, School of Dental Science, University of Melbourne. Copyright 9 1995 by Mosby-Year Book, Inc. 1079-2104/95/$3.00 + 0 7/14/61723
Fig. 1. Case 1. Radiograph illustrates two embedded teeth with circumcoronal radiolucencies. fibrillar tufts peripherally, which resembled Sharpey's fibers. Case 2 This 40-year-old man had seven deeply embedded teeth that exhibited circumcoronal radiolucencies containing irregular radiopacities (Fig. 6). Microscopic findings, The lesions exhibited a similar appearance to those of Case 1 except that the Type B calcifications were larger and much more numerous; Type A calcifications were evident in only some of the Type B calcifications. DISCUSSION The clinical features of the four reported cases of this condition are summarized in Table I. The microscopic findings in the present two cases are similar to those in the two reported previously. 1, 2 We have had the opportunity to study the pathology sections of the example reported by Sandler et al. 1 These were very similar to those of our Case 1; the Type A calcifications and whorled structures were numerous, but 603
604
Gardner and Radden
ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGY
May 1995
Fig. 2. Case 1. Low-power photomicrograph illustrates whorled structures and Type A calcifications within fibrous connective tissue stroma. No rests of odontogenic epithelium are apparent in this field, although they were present elsewhere. (Hematoxylin-eosin stain; original magnification X62.5.)
Fig. 3. Case 1. Higher magnification of part of the field illustrated in Fig. 2 shows Type A calcifications in more detail. They do not polarize. (Hematoxylin-eosin stain; original magnification X125.) there was no evidence of Type B calcifications. The case reported by Lukinmaa et al. 2 exhibited Type A calcifications only. The histologic appearance of these follicles is identical to that of the hyperplastic dental follicles of regional odontodysplasia 4, 5 although the clinical findings do not support that diagnosis. These findings of rests of odontogenic epithelium, whorled structures, and Type A and B calcifications are characteristic of regional odontodysplasia but have also been observed
in the calcifying epithelial odontogenic tumor, 4 the follicles of unerupted teeth in amelogenesis imperfecta and dens in dente, 6 and adjacent to enamel pearls.4, 7 The histologic resemblance of these lesions to the variant of odontogenic fibroma that has been referred to as the W H O type 3 has been stressed by Sandier et al. 1 and by Lukinmaa et al. 2 This suggestion is not surprising in that the odontogenic fibroma ( W H O type) also exhibits calcifications and rests of odonto-
ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGY Volume 79, Number 5
Gardner and Radden
605
Fig. 4. Case 2. Type B calcification incorporating few Type A calcifications. Type A calcifications are also present at bottom of illustration. (Hematoxylin-eosin stain; original magnification X62.5.)
Fig. 5. Case 2. Part of Type B calcification illustrated in Fig. 4 photographed under polarized light. Type B calcification polarizes, illustrating its fibrillar structure, while the Type A calcifications incorporated within the Type B calcification do not. (Hematoxylin-eosin stain; original magnification X125.) genic epithelium; it may well be derived from the dental follicle. However, there are differences. A fundamental component of the odontogenic fibroma ( W H O type) is that the connective tissue is very cellular, consisting of plump fibroblasts, and could therefore be described as fibroblastic. Moreover, this cellular connective tissUe is often interwoven with less cellular areas and may be quite vascular. 8 This type of connective tissue is not a feature of the present lesions. Moreover, the odontogenic fibroma ( W H O
type) often exhibits material that could be considered dysplastic dentin or osteoid; this material has not so far been reported in the present lesions. We c o n c l u d e that they resemble the hyperplastic follicles of regional odontodysplasia and indeed are identical to them. The cause of this condition is not known. It represents multiple hyperplastic dental follicles containing calcified tissue similar to that found in the hyperplastic follicles of regional odontodysplasia but without
606
Gardner and Radden
ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGY
May1995
relationship of the present condition to either cleidocranial dysplasia or to the Gardner syndrome, two other disorders in which multiple unerupted teeth occur. The present rare condition is sufficiently distinctive to be considered a pathologic entity for which we suggest the term, multiple calcifying hyperplastic
dental follicles. We thank Dr. Leon Eisenbud for providing our Case 1 and Dr. Edmund Cataldo and his colleagues for providing pathology sections of their case. 2
Fig. 6. Case 2. Radiograph illustrates seven embedded teeth with circumcoronal radiolucencies. Radiolucencies in left mandible demonstrate radiopacities best. Table I. Cases of multiple calcifying hyperplastic
dental follicles Reports
Patient
Teeth affected
Sandier et al. 1 1988 Lukinmaa et al. 2 1990
15-year-old male
13 unerupted teeth Left mandibular second premolar and second molar Left mandibular second and third molars 7 unerupted teeth
Gardner and Radden 1995 Case 1 Gardner and Radden 1995 Case 2
24-year-old male
26-year-old white male 40-year-old male
REFERENCES 1. Sandier H J, Nersasian RR, Cataldo E, Pochebit S, Dayal Y. Multiple dental follicles with odontogenic fibroma-like changes (WHO type). ORAL SURG ORAL MED ORAL PATHOL 1988; 66:78-84. 2. Lukinmaa P-L, Hietanen J, Anttinen J, Ahonen P. Contiguous enlarged dental follicles with histologic features resembling the WHO type of odontogenic fibroma. ORAL SURG ORAL MED ORAL PATHOL 1990;70:313-7. 3. Gardner DG. The central odontogenic fibroma: an attempt at clarification. ORAL SURG ORAL MED ORAL PATHOL 1980; 50:425-32. 4. Gardner DG, Sapp JP. Regional odontodysplasia. ORAL SURG ORAL MED ORAL PATHOL 1973;35:351-65. 5. Crawford PJM, Aldred MJ. Regional odontodysplasia: a bibliography. J Oral Pathol Med 1989;18:251-63. 6. Witkop C J, Sauk JJ. Dental and oral manifestations of hereditary disease. Washington, DC: American Academy of Oral Pathology, 1971:29-30. 7. Pindborg JJ. Pathology of the dental hard tissues. Philadelphia: WB Saunders, 1970:120-3. 8. Gardner DG. The peripheral odontogenic fibroma: an attempt at clarification. ORAL SURG ORAL MED ORAL PATHOL 1982; 54:40-8.
Reprint requests:
the malformations of the teeth in that condition. In turn, these hyperplastic follicles probably prevent the eruption of the involved teeth, although this possibility cannot be proved. Moreover, there is no apparent
David G. Gardner, DDS, MSD University of Colorado School of Dentistry Campus Box C284 4200 East Ninth Avenue Denver, CO 80262
This article describes two additional examples of this unusual condition; two others were reported previously. The microscopic features are those of the hyperplastic dental follicles that occur in regional odontodysplasia. However, the teeth are not defective, as in regional odontodysplasia. This rare condition is sufficiently distinctive to be considered a pathologic entity. (ORAL SURGORALMED ORALPATHOLORALRADIOL[NDOD 1995;79:603-6)
To date, two examples have been published 1, 2 of enlarged dental follicles that were associated with multiple unerupted teeth and that exhibited abundant calcifications and rests of odontogenic epithelium. In both cases, the authors considered them to be hamartomas and likened their histologic appearance to that of the central odontogenic fibroma ( W H O type) 3 and of the hyperplastic follicles of regional odontodysplasia. 4 The purpose of the present article is to report two additional examples and to discuss the nature of this unusual condition. CASE REPORTS Case 1 This patient was a 26-year-old white man whose mandibular left second and third molar teeth had not erupted (Fig. 1). Both teeth exhibited circumcoronal radiolucencies that were approximately 6 mm wide and that were found at surgery to consist of dense fibrous connective tissue. Microscopic findings.The lesions consisted of relatively dense, moderately cellular, fibrous connective tissue, throughout which were scattered numerous rests of odontogenic epithelium and whorled structures composed of fibroblasts, collagen fibers arranged concentrically, and small calcifications similar to those that have been described as Type "A" calcifications in the hyperplastic follicles of regional odontodysplasia (Figs. 2 and 3). In many areas these calcifications had coalesced into larger bodies in which the original small calcifications could be clearly identified. In a few areas the whorled structures exhibited only granular calcifications. Elsewhere, the Type "A" calcifications, which did not polarize, had become incorporated into an eosinophilic material (Fig. 4), the fibrillar nature of which was demonstrable by polarization microscopy (Fig. 5). These Type "B" calcifications (Figs. 4 and 5) also exhibited aprofessor, Divisionof Oral Pathologyand Oncology,Universityof Colorado School of Dentistry. bSenior Associate, School of Dental Science, University of Melbourne. Copyright 9 1995 by Mosby-Year Book, Inc. 1079-2104/95/$3.00 + 0 7/14/61723
Fig. 1. Case 1. Radiograph illustrates two embedded teeth with circumcoronal radiolucencies. fibrillar tufts peripherally, which resembled Sharpey's fibers. Case 2 This 40-year-old man had seven deeply embedded teeth that exhibited circumcoronal radiolucencies containing irregular radiopacities (Fig. 6). Microscopic findings, The lesions exhibited a similar appearance to those of Case 1 except that the Type B calcifications were larger and much more numerous; Type A calcifications were evident in only some of the Type B calcifications. DISCUSSION The clinical features of the four reported cases of this condition are summarized in Table I. The microscopic findings in the present two cases are similar to those in the two reported previously. 1, 2 We have had the opportunity to study the pathology sections of the example reported by Sandler et al. 1 These were very similar to those of our Case 1; the Type A calcifications and whorled structures were numerous, but 603
604
Gardner and Radden
ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGY
May 1995
Fig. 2. Case 1. Low-power photomicrograph illustrates whorled structures and Type A calcifications within fibrous connective tissue stroma. No rests of odontogenic epithelium are apparent in this field, although they were present elsewhere. (Hematoxylin-eosin stain; original magnification X62.5.)
Fig. 3. Case 1. Higher magnification of part of the field illustrated in Fig. 2 shows Type A calcifications in more detail. They do not polarize. (Hematoxylin-eosin stain; original magnification X125.) there was no evidence of Type B calcifications. The case reported by Lukinmaa et al. 2 exhibited Type A calcifications only. The histologic appearance of these follicles is identical to that of the hyperplastic dental follicles of regional odontodysplasia 4, 5 although the clinical findings do not support that diagnosis. These findings of rests of odontogenic epithelium, whorled structures, and Type A and B calcifications are characteristic of regional odontodysplasia but have also been observed
in the calcifying epithelial odontogenic tumor, 4 the follicles of unerupted teeth in amelogenesis imperfecta and dens in dente, 6 and adjacent to enamel pearls.4, 7 The histologic resemblance of these lesions to the variant of odontogenic fibroma that has been referred to as the W H O type 3 has been stressed by Sandier et al. 1 and by Lukinmaa et al. 2 This suggestion is not surprising in that the odontogenic fibroma ( W H O type) also exhibits calcifications and rests of odonto-
ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGY Volume 79, Number 5
Gardner and Radden
605
Fig. 4. Case 2. Type B calcification incorporating few Type A calcifications. Type A calcifications are also present at bottom of illustration. (Hematoxylin-eosin stain; original magnification X62.5.)
Fig. 5. Case 2. Part of Type B calcification illustrated in Fig. 4 photographed under polarized light. Type B calcification polarizes, illustrating its fibrillar structure, while the Type A calcifications incorporated within the Type B calcification do not. (Hematoxylin-eosin stain; original magnification X125.) genic epithelium; it may well be derived from the dental follicle. However, there are differences. A fundamental component of the odontogenic fibroma ( W H O type) is that the connective tissue is very cellular, consisting of plump fibroblasts, and could therefore be described as fibroblastic. Moreover, this cellular connective tissUe is often interwoven with less cellular areas and may be quite vascular. 8 This type of connective tissue is not a feature of the present lesions. Moreover, the odontogenic fibroma ( W H O
type) often exhibits material that could be considered dysplastic dentin or osteoid; this material has not so far been reported in the present lesions. We c o n c l u d e that they resemble the hyperplastic follicles of regional odontodysplasia and indeed are identical to them. The cause of this condition is not known. It represents multiple hyperplastic dental follicles containing calcified tissue similar to that found in the hyperplastic follicles of regional odontodysplasia but without
606
Gardner and Radden
ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGY
May1995
relationship of the present condition to either cleidocranial dysplasia or to the Gardner syndrome, two other disorders in which multiple unerupted teeth occur. The present rare condition is sufficiently distinctive to be considered a pathologic entity for which we suggest the term, multiple calcifying hyperplastic
dental follicles. We thank Dr. Leon Eisenbud for providing our Case 1 and Dr. Edmund Cataldo and his colleagues for providing pathology sections of their case. 2
Fig. 6. Case 2. Radiograph illustrates seven embedded teeth with circumcoronal radiolucencies. Radiolucencies in left mandible demonstrate radiopacities best. Table I. Cases of multiple calcifying hyperplastic
dental follicles Reports
Patient
Teeth affected
Sandier et al. 1 1988 Lukinmaa et al. 2 1990
15-year-old male
13 unerupted teeth Left mandibular second premolar and second molar Left mandibular second and third molars 7 unerupted teeth
Gardner and Radden 1995 Case 1 Gardner and Radden 1995 Case 2
24-year-old male
26-year-old white male 40-year-old male
REFERENCES 1. Sandier H J, Nersasian RR, Cataldo E, Pochebit S, Dayal Y. Multiple dental follicles with odontogenic fibroma-like changes (WHO type). ORAL SURG ORAL MED ORAL PATHOL 1988; 66:78-84. 2. Lukinmaa P-L, Hietanen J, Anttinen J, Ahonen P. Contiguous enlarged dental follicles with histologic features resembling the WHO type of odontogenic fibroma. ORAL SURG ORAL MED ORAL PATHOL 1990;70:313-7. 3. Gardner DG. The central odontogenic fibroma: an attempt at clarification. ORAL SURG ORAL MED ORAL PATHOL 1980; 50:425-32. 4. Gardner DG, Sapp JP. Regional odontodysplasia. ORAL SURG ORAL MED ORAL PATHOL 1973;35:351-65. 5. Crawford PJM, Aldred MJ. Regional odontodysplasia: a bibliography. J Oral Pathol Med 1989;18:251-63. 6. Witkop C J, Sauk JJ. Dental and oral manifestations of hereditary disease. Washington, DC: American Academy of Oral Pathology, 1971:29-30. 7. Pindborg JJ. Pathology of the dental hard tissues. Philadelphia: WB Saunders, 1970:120-3. 8. Gardner DG. The peripheral odontogenic fibroma: an attempt at clarification. ORAL SURG ORAL MED ORAL PATHOL 1982; 54:40-8.
Reprint requests:
the malformations of the teeth in that condition. In turn, these hyperplastic follicles probably prevent the eruption of the involved teeth, although this possibility cannot be proved. Moreover, there is no apparent
David G. Gardner, DDS, MSD University of Colorado School of Dentistry Campus Box C284 4200 East Ninth Avenue Denver, CO 80262