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Cholesterol granuloma of the maxilla
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CASE REPORT
Cholesterol granuloma of the maxilla Granulome à cholestérol du maxillaire N. Baldini a, A. Pare a, S. Badja a, A. Veyssiere a, J.D. Kün-Darbois a,b,∗ a
Service de chirurgie maxillo-faciale et stomatologie, CHU d’Angers, 4, rue Larrey, 49933 Angers cedex, France b Groupe études remodelage osseux et biomatériaux (GEROM) — LHEA, IRIS-Institut de biologie en santé (IBS), université d’Angers, CHU d’Angers, 49933 Angers cedex, France
KEYWORDS Cholesterol granuloma; Cholesterol cyst; Jaws; Maxilla
Summary Intra-osseous cholesterol granuloma (CG) is a rare and benign lesion. Very few cases of CG of the jaws have been described in the literature. CG of the jaws seems to be due to the accumulation of cholesterol of hematogenous origin in odontogenic cysts. We report on one case of CG of the maxilla treated by surgical enucleation in a 46-year-old man who presented an asymptomatic swelling of the maxilla. © 2018 Elsevier Masson SAS. All rights reserved.
Résumé Le granulome à cholestérol (GC) intra-osseux est une lésion rare et bénigne. Très peu de cas de GC des mâchoires ont été décrits à ce jour dans la littérature. Les GC des maxillaires semblent être dus à l’accumulation de cholestérol d’origine hématogène dans des kystes odontogènes. Nous rapportons un cas de GC du maxillaire traité par énucléation chirurgicale chez un homme de 46 ans qui présentait une tuméfaction maxillaire asymptomatique. © 2018 Elsevier Masson SAS. Tous droits r´ eserv´ es.
∗ Corresponding author at: Groupe études remodelage osseux et biomatériaux (GEROM) — LHEA, IRIS-Institut de biologie en santé (IBS), université d’Angers, CHU d’Angers, 49933 Angers cedex, France. E-mail address: [email protected] (J.D. Kün-Darbois).
https://doi.org/10.1016/j.morpho.2018.01.001 1286-0115/© 2018 Elsevier Masson SAS. All rights reserved.
Please cite this article in press as: Baldini N, et al. Cholesterol granuloma of the maxilla. Morphologie (2018), https://doi.org/10.1016/j.morpho.2018.01.001
+Model MORPHO-339; No. of Pages 3
ARTICLE IN PRESS
2
N. Baldini et al.
Introduction Intra-osseous cholesterol granuloma (CG) is a rare and benign lesion. CG is considered as an inflammatory reaction to the deposition of cholesterol crystals inside a bone [1,2]. Histological analyses report that CG are made of a fibrous granulation tissue filled with rhomboid shaped-cleft cholesterol crystals, surrounded by giant cells and macrophages associated with hemosiderin [1,3]. They are most commonly observed at the middle ear, the petrous apex, the maxillary sinus or the orbit [4—6]. Very few cases of CG of the jaws (maxilla or mandible) have been reported to date [1—3,7—12]. We report on one case of CG of the maxilla that occurred in a residual radicular cyst.
Case report A 46-year-old man presented with an asymptomatic chronic maxillary swelling. The lesion had been noticed by the patient himself for the first time about 4 months prior. Clinical examination revealed a bony swelling of the maxilla in an area where the tooth 26 had been removed two years before because of an infection. There was no fistula, no pain and no sign of local infection. The adjacent teeth (25 and 27) responded positively to thermal pulp testing performed with CO2 snow. The panoramic radiograph showed a well-delineated, ovoid radiolucent lesion, 14 mm in diameter localized on the left part of the maxilla between the roots of teeth 25 and 27 and under the left maxillary sinus (Fig. 1). Computed tomography (CT) showed that the lesion was independent from the maxillary sinus and the adjacent teeth (Fig. 2).
Figure 1
The diagnosis of residual radicular cyst was evoked. Surgical enucleation of the cyst was performed using an intra-oral approach (via a mucosal incision, a sub-periosteal detachment and the use of a surgical drill with a round bur). The lesion was sent for histopathological analysis after fixation in 10% formalin. Wound healing was obtained a few days after the procedure. A lipid profile was performed and showed normal blood rates of low-density lipoprotein, highdensity lipoprotein, triglycerides and total cholesterol. The post-operative follow up was uneventful with no signs of recurrence or complication after 1 year. Histological examination revealed a non-keratinized stratified squamous cystic epithelial wall. The cyst contained spindle shaped voids characteristic of cholesterol crystals. However, the processing of the tissues had involved organic solvents such as xylene that had dissolved the lipids and cholesterol. Each cholesterol phantom was surrounded by a fibrous stroma containing giant cells and macrophages with hemosiderin pigments (Fig. 3).
Discussion Although cases of periapical cysts containing some cholesterol crystals are reported, there are few cases of cholesterol granulomas of the jaws described to date [1—4,7—9,11,12]. It is likely that this benign lesion is under reported. Intra-osseous CG is mostly found in edentulous spaces of the jaws as in the present case [1,3]. CG seems to be due to the accumulation of cholesterol of hematogenous origin in any type of odontogenic cysts [9,12]. The origin of cholesterol crystals accumulation is not clearly elucidated yet. Trauma has been advocated via the
Panoramic radiograph showing an ovoid radiolucent lesion in the region of the missing upper left first molar (26) (arrows).
Figure 2 CT scan focused on the maxilla in frontal view (A) and frontal reconstruction (B) showing a cystic lesion between the teeth 25 and 27.
Please cite this article in press as: Baldini N, et al. Cholesterol granuloma of the maxilla. Morphologie (2018), https://doi.org/10.1016/j.morpho.2018.01.001
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Cholesterol granuloma of the maxilla
3 be due to the accumulation of cholesterol caused by the obstruction of the Eustachian tube [1]. Although surgical enucleation is the usual adequate treatment, Lee et al., reported one case of large CG of the mandible treated by hemimandibulectomy and free flap reconstruction [2]. No case of infectious complications or malignant transformation has been described to our knowledge. The authors recommend to perform a systematic surgical enucleation as soon as the diagnosis of such lesion is done in order to avoid growth-related complications. The diagnosis must be confirmed by histological examination.
Disclosure of interest The authors declare that they have no competing interest.
References
Figure 3 Histological analysis of the cyst. A) Low magnification of the cyst showing the capsule and the fibrous tissue containing phantoms of cholesterol crystals appearing in white due to their dissolution by the histologic solvents. B). High magnification of the cyst: the crystal phantoms are outlined by multinucleated giant cells (arrows), some macrophages containing hemosiderin pigments appear in brown (arrowhead) hematoxylin, phloxin, saffron staining.
formation of a hematoma due to poor drainage secondarily leading to the accumulation of cholesterol [3]. Crystals of cholesterol have also been related to the disintegration of red blood cells, plasma lipids or fatty degeneration of connective tissue. Another possible origin for CG in pre-existing jaw cysts has been suggested by Yamazaki et al.; CG could be due to the presence of basement membrane-type heparin sulfate proteoglycan trapping low-density lipoprotein in the cyst wall [10]. On the other hand, CG localized in other bones (i.e. orbit, middle ear, petrous apex) does not share the same mechanisms of disease since no odontogenic cysts are localized there. The origin of CG in the middle ear seems to
[1] Hirshberg A, Dayan D, Buchner A, Freedman A. Cholesterol granuloma of the jaws. Report of a case. Int J Oral Maxillofac Surg 1988;17:230—1. [2] Lee J, Alrashdan M, Ahn K, Kang M, Hong S, Kim S. Cholesterol granuloma in the wall of a mandibular dentigerous cyst: a rare case report. J Clin Exp Dent 2010;2:e88—90. [3] Fernández-Olarte H. An unusual case of cholesterol granuloma of the jaws. EC Dent Sci 2017;12:83—6. [4] Hernandez-Estrada RA, Kshettry VR, Vogel AN, Curtis MT, Evans JJ. Cholesterol granulomas presenting as sellar masses: a similar, but clinically distinct entity from craniopharyngioma and Rathke’s cleft cyst. Pituitary 2017;20:325—32. [5] Jang CH, Kim YH, Cho YB. A primary large cholesterol cyst of the mastoid presenting with dysgeusia. In Vivo 2009;23:167—9. [6] Fukuta K, Jackson IT. Epidermoid cyst and cholesterol granuloma of the orbit. Br J Plast Surg 1990;43:521—7. [7] Kamboj M, Devi A, Gupta S. Cholesterol granuloma in odontogenic cyst: an enigmatic lesion. Case Rep Dent 2016;2016:6105142. [8] Alkan A, Etoz O, Candirli C, Ulu M, Dayisoylu EH. Cholesterol granuloma of the jaws: report of two cases. J Pak Med Assoc 2014;64:86—8. [9] Aparna M, Gupta M, Sujir N, Kamath A, Solomon M, Pai K, et al. Calcifying odontogenic cyst: a rare report of a nonneoplastic variant associated with cholesterol granuloma. J Contemp Dent Pract 2013;14:1178—82. [10] Yamazaki M, Cheng J, Hao N, Takagi R, Jimi S, Itabe H, et al. Basement membrane-type heparan sulfate proteoglycan (perlecan) and low-density lipoprotein (LDL) are co-localized in granulation tissues: a possible pathogenesis of cholesterol granulomas in jaw cysts. J Oral Pathol Med 2004;33:177—84. [11] Kaffe I, Littner MM, Buchner A, Friedman M. Cholesterol granuloma embedded in an odontoma of the maxilla. J Oral Maxillofac Surg 1984;42:319—22. [12] Browne R. The origin of cholesterol in odontogenic cysts in man. Archives of oral biology 1971;16:107IN21—IN113.
Please cite this article in press as: Baldini N, et al. Cholesterol granuloma of the maxilla. Morphologie (2018), https://doi.org/10.1016/j.morpho.2018.01.001
ARTICLE IN PRESS
MORPHO-339; No. of Pages 3 Morphologie (2018) xxx, xxx—xxx
Disponible en ligne sur
ScienceDirect www.sciencedirect.com
CASE REPORT
Cholesterol granuloma of the maxilla Granulome à cholestérol du maxillaire N. Baldini a, A. Pare a, S. Badja a, A. Veyssiere a, J.D. Kün-Darbois a,b,∗ a
Service de chirurgie maxillo-faciale et stomatologie, CHU d’Angers, 4, rue Larrey, 49933 Angers cedex, France b Groupe études remodelage osseux et biomatériaux (GEROM) — LHEA, IRIS-Institut de biologie en santé (IBS), université d’Angers, CHU d’Angers, 49933 Angers cedex, France
KEYWORDS Cholesterol granuloma; Cholesterol cyst; Jaws; Maxilla
Summary Intra-osseous cholesterol granuloma (CG) is a rare and benign lesion. Very few cases of CG of the jaws have been described in the literature. CG of the jaws seems to be due to the accumulation of cholesterol of hematogenous origin in odontogenic cysts. We report on one case of CG of the maxilla treated by surgical enucleation in a 46-year-old man who presented an asymptomatic swelling of the maxilla. © 2018 Elsevier Masson SAS. All rights reserved.
Résumé Le granulome à cholestérol (GC) intra-osseux est une lésion rare et bénigne. Très peu de cas de GC des mâchoires ont été décrits à ce jour dans la littérature. Les GC des maxillaires semblent être dus à l’accumulation de cholestérol d’origine hématogène dans des kystes odontogènes. Nous rapportons un cas de GC du maxillaire traité par énucléation chirurgicale chez un homme de 46 ans qui présentait une tuméfaction maxillaire asymptomatique. © 2018 Elsevier Masson SAS. Tous droits r´ eserv´ es.
∗ Corresponding author at: Groupe études remodelage osseux et biomatériaux (GEROM) — LHEA, IRIS-Institut de biologie en santé (IBS), université d’Angers, CHU d’Angers, 49933 Angers cedex, France. E-mail address: [email protected] (J.D. Kün-Darbois).
https://doi.org/10.1016/j.morpho.2018.01.001 1286-0115/© 2018 Elsevier Masson SAS. All rights reserved.
Please cite this article in press as: Baldini N, et al. Cholesterol granuloma of the maxilla. Morphologie (2018), https://doi.org/10.1016/j.morpho.2018.01.001
+Model MORPHO-339; No. of Pages 3
ARTICLE IN PRESS
2
N. Baldini et al.
Introduction Intra-osseous cholesterol granuloma (CG) is a rare and benign lesion. CG is considered as an inflammatory reaction to the deposition of cholesterol crystals inside a bone [1,2]. Histological analyses report that CG are made of a fibrous granulation tissue filled with rhomboid shaped-cleft cholesterol crystals, surrounded by giant cells and macrophages associated with hemosiderin [1,3]. They are most commonly observed at the middle ear, the petrous apex, the maxillary sinus or the orbit [4—6]. Very few cases of CG of the jaws (maxilla or mandible) have been reported to date [1—3,7—12]. We report on one case of CG of the maxilla that occurred in a residual radicular cyst.
Case report A 46-year-old man presented with an asymptomatic chronic maxillary swelling. The lesion had been noticed by the patient himself for the first time about 4 months prior. Clinical examination revealed a bony swelling of the maxilla in an area where the tooth 26 had been removed two years before because of an infection. There was no fistula, no pain and no sign of local infection. The adjacent teeth (25 and 27) responded positively to thermal pulp testing performed with CO2 snow. The panoramic radiograph showed a well-delineated, ovoid radiolucent lesion, 14 mm in diameter localized on the left part of the maxilla between the roots of teeth 25 and 27 and under the left maxillary sinus (Fig. 1). Computed tomography (CT) showed that the lesion was independent from the maxillary sinus and the adjacent teeth (Fig. 2).
Figure 1
The diagnosis of residual radicular cyst was evoked. Surgical enucleation of the cyst was performed using an intra-oral approach (via a mucosal incision, a sub-periosteal detachment and the use of a surgical drill with a round bur). The lesion was sent for histopathological analysis after fixation in 10% formalin. Wound healing was obtained a few days after the procedure. A lipid profile was performed and showed normal blood rates of low-density lipoprotein, highdensity lipoprotein, triglycerides and total cholesterol. The post-operative follow up was uneventful with no signs of recurrence or complication after 1 year. Histological examination revealed a non-keratinized stratified squamous cystic epithelial wall. The cyst contained spindle shaped voids characteristic of cholesterol crystals. However, the processing of the tissues had involved organic solvents such as xylene that had dissolved the lipids and cholesterol. Each cholesterol phantom was surrounded by a fibrous stroma containing giant cells and macrophages with hemosiderin pigments (Fig. 3).
Discussion Although cases of periapical cysts containing some cholesterol crystals are reported, there are few cases of cholesterol granulomas of the jaws described to date [1—4,7—9,11,12]. It is likely that this benign lesion is under reported. Intra-osseous CG is mostly found in edentulous spaces of the jaws as in the present case [1,3]. CG seems to be due to the accumulation of cholesterol of hematogenous origin in any type of odontogenic cysts [9,12]. The origin of cholesterol crystals accumulation is not clearly elucidated yet. Trauma has been advocated via the
Panoramic radiograph showing an ovoid radiolucent lesion in the region of the missing upper left first molar (26) (arrows).
Figure 2 CT scan focused on the maxilla in frontal view (A) and frontal reconstruction (B) showing a cystic lesion between the teeth 25 and 27.
Please cite this article in press as: Baldini N, et al. Cholesterol granuloma of the maxilla. Morphologie (2018), https://doi.org/10.1016/j.morpho.2018.01.001
+Model MORPHO-339; No. of Pages 3
ARTICLE IN PRESS
Cholesterol granuloma of the maxilla
3 be due to the accumulation of cholesterol caused by the obstruction of the Eustachian tube [1]. Although surgical enucleation is the usual adequate treatment, Lee et al., reported one case of large CG of the mandible treated by hemimandibulectomy and free flap reconstruction [2]. No case of infectious complications or malignant transformation has been described to our knowledge. The authors recommend to perform a systematic surgical enucleation as soon as the diagnosis of such lesion is done in order to avoid growth-related complications. The diagnosis must be confirmed by histological examination.
Disclosure of interest The authors declare that they have no competing interest.
References
Figure 3 Histological analysis of the cyst. A) Low magnification of the cyst showing the capsule and the fibrous tissue containing phantoms of cholesterol crystals appearing in white due to their dissolution by the histologic solvents. B). High magnification of the cyst: the crystal phantoms are outlined by multinucleated giant cells (arrows), some macrophages containing hemosiderin pigments appear in brown (arrowhead) hematoxylin, phloxin, saffron staining.
formation of a hematoma due to poor drainage secondarily leading to the accumulation of cholesterol [3]. Crystals of cholesterol have also been related to the disintegration of red blood cells, plasma lipids or fatty degeneration of connective tissue. Another possible origin for CG in pre-existing jaw cysts has been suggested by Yamazaki et al.; CG could be due to the presence of basement membrane-type heparin sulfate proteoglycan trapping low-density lipoprotein in the cyst wall [10]. On the other hand, CG localized in other bones (i.e. orbit, middle ear, petrous apex) does not share the same mechanisms of disease since no odontogenic cysts are localized there. The origin of CG in the middle ear seems to
[1] Hirshberg A, Dayan D, Buchner A, Freedman A. Cholesterol granuloma of the jaws. Report of a case. Int J Oral Maxillofac Surg 1988;17:230—1. [2] Lee J, Alrashdan M, Ahn K, Kang M, Hong S, Kim S. Cholesterol granuloma in the wall of a mandibular dentigerous cyst: a rare case report. J Clin Exp Dent 2010;2:e88—90. [3] Fernández-Olarte H. An unusual case of cholesterol granuloma of the jaws. EC Dent Sci 2017;12:83—6. [4] Hernandez-Estrada RA, Kshettry VR, Vogel AN, Curtis MT, Evans JJ. Cholesterol granulomas presenting as sellar masses: a similar, but clinically distinct entity from craniopharyngioma and Rathke’s cleft cyst. Pituitary 2017;20:325—32. [5] Jang CH, Kim YH, Cho YB. A primary large cholesterol cyst of the mastoid presenting with dysgeusia. In Vivo 2009;23:167—9. [6] Fukuta K, Jackson IT. Epidermoid cyst and cholesterol granuloma of the orbit. Br J Plast Surg 1990;43:521—7. [7] Kamboj M, Devi A, Gupta S. Cholesterol granuloma in odontogenic cyst: an enigmatic lesion. Case Rep Dent 2016;2016:6105142. [8] Alkan A, Etoz O, Candirli C, Ulu M, Dayisoylu EH. Cholesterol granuloma of the jaws: report of two cases. J Pak Med Assoc 2014;64:86—8. [9] Aparna M, Gupta M, Sujir N, Kamath A, Solomon M, Pai K, et al. Calcifying odontogenic cyst: a rare report of a nonneoplastic variant associated with cholesterol granuloma. J Contemp Dent Pract 2013;14:1178—82. [10] Yamazaki M, Cheng J, Hao N, Takagi R, Jimi S, Itabe H, et al. Basement membrane-type heparan sulfate proteoglycan (perlecan) and low-density lipoprotein (LDL) are co-localized in granulation tissues: a possible pathogenesis of cholesterol granulomas in jaw cysts. J Oral Pathol Med 2004;33:177—84. [11] Kaffe I, Littner MM, Buchner A, Friedman M. Cholesterol granuloma embedded in an odontoma of the maxilla. J Oral Maxillofac Surg 1984;42:319—22. [12] Browne R. The origin of cholesterol in odontogenic cysts in man. Archives of oral biology 1971;16:107IN21—IN113.
Please cite this article in press as: Baldini N, et al. Cholesterol granuloma of the maxilla. Morphologie (2018), https://doi.org/10.1016/j.morpho.2018.01.001