대한소아치과학회지 (Journal of the korean academy of Pediatric Dentistry)

  • 제36권3호
  • /
  • Pages.489-497
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  • 2009
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  • 1226-8496(pISSN)
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  • 2288-3819(eISSN)
대한소아치과학회 (Korean Academy of Pediatric Dentistry)
권호 표지

소아에서 발생한 악골의 다발성 각화낭성 치성종양의 치험례

TREATMENT OF MULTIPLE JAW KERATOCYSTIC ODONTOGENIC TUMOR IN CHILDREN'S JAW BONE : A CASE REPORT

  • 김지영 (경북대학교 치과대학 소아치과학교실) ;
  • 김영진 (경북대학교 치과대학 소아치과학교실) ;
  • 김현정 (경북대학교 치과대학 소아치과학교실) ;
  • 남순현 (경북대학교 치과대학 소아치과학교실)
  • Kim, Ji-Young (Pediatric dentistry, School of Dentistry, Kyungpook National University) ;
  • Kim, Young-Jin (Pediatric dentistry, School of Dentistry, Kyungpook National University) ;
  • Kim, Hyun-Jung (Pediatric dentistry, School of Dentistry, Kyungpook National University) ;
  • Nam, Soon-Hyeun (Pediatric dentistry, School of Dentistry, Kyungpook National University)
  • 발행 : 2009.08.31
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초록

악골에서 다수로 발생하는 낭은, 단독으로 발생하는 낭에 비해 매우 드물게 나타난다. 증후군을 동반하지는 않고, 악골 내에 다수의 낭이 발생한 경우를 다발성 악골낭 이라고 하는데, 소아에서 발생하는 다발성 악골 낭의 대부분은 각화낭성 치성종양(Keratocystic odontogenic tumor, KCOT)이다. 소아에서 나타나는 악골의 다발성 각화낭성 치성종양은, 기저세포모반증후군의 한 증상으로 발생할 수 있으므로, 악골의 방사선 사진에서 다발성 낭이 발견되면, 증후군을 의심해보고 임상 및 병리 검사를 하게 된다. 본 증례는, 다발성 악골 낭을 주소로 내원한 소아환자들로, 기저세포모반증후군을 의심하였으나 현재까지 증후군의 다른 증상들은 보이고 있지 않으며, 단지 수년간에 걸쳐서 종양의 재발과 수술을 반복하고 있는 경우이다. 악골의 다발성 낭 이외의 기저세포모반증후군의 증상들이 나타나지 않더라도, 나이가 들면 증후군의 다른 증상들이 나타난 경우의 보고도 있으므로, 소아에서 이러한 다발성 치성종양이 관찰되면, 지속적으로 방사선 및 임상 검사를 시행하여 증후군으로의 진행 여부를 관찰하는 것이 중요한 것으로 사료된다.

Occurrence of multiple cysts in jaw bone is rare compared to solitary cysts. numerous cysts occurring in jaw bone which not accompany any syndromes are defined as multiple jaw cysts, and most of these cases in children are keratocystic odontogenic tumor (KCOT) Multiple KCOT occurring in children are often associated with basal cell nevus syndrome(BCNS), so if multiple cysts are found on the radiograph, we suspect this syndrome and pursue clinical and pathological tests. In this case, a pediatric patient, reporting with multiple cysts in the jaw was suspected of BCNS, but hasn't shown any other symptoms of this syndrome up to date, and has kept repeating surgical operation and recurrence of the tumor. Although no symptoms besides multiple jaw cysts is present, it is often reported that other symptoms appear late in the patient's age. Therefore, in cases where multiple odontogenic tumors are found in children, continuous radiographic and clinical follow-ups in order to check the progress of the syndrome is considered important.

키워드

참고문헌

  1. Chiang ML, Huang WH : Odontogenic keratocyst clinically mimicking an eruption cyst : report of a case. J Oral Pathol Med, 33:373-375, 2004. https://doi.org/10.1111/j.1600-0714.2004.00135.x
  2. Shear M, Speight PM : Cysts of the oral and maxillofacial regions, Blackwell Munksgaard, fourth edition: 6-58, 2007.
  3. Ortega A, Garcia O, Zepeda S, et al. : Gorlin-Goltz syndrome : clinicopathologic aspects. Med Oral Patol Oral Cir Bucal, 13:E338-343, 2008.
  4. Stoll C, Stollenwerk C, Riediger D, et al. : Cytokeratin expression patterns for distinction of odontogenic keratocysts from dentigerous and radicular cysts. J Oral Pathol Med, 34:558-564, 2005. https://doi.org/10.1111/j.1600-0714.2005.00352.x
  5. Woolgar JA, Rippin JW, Browne RM : The odontogenic keratocyst and its occurrence in the nevoid basal cell carcinoma syndrome. Oral Surg Oral Med Oral Pathol, 64:727-730, 1987. https://doi.org/10.1016/0030-4220(87)90176-9
  6. Auluck A, Suhas S, Pai KM : Multiple odontogenic keratocysts : report of a case. J Can Dent Assoc, 72:651-656, 2006.
  7. White JC : Multiple benign cystic epitheliomas. J cutaneous and genitourinary disease, 12:477-484, 1894.
  8. Gorlin RJ, Goltz RW : Multiple nevoid basal-cell epithelioma, jaw cysts, and bifid rib syndromes. New england J of medicine, 262:908-912, 1960. https://doi.org/10.1056/NEJM196005052621803
  9. Anderson DE, Howell JB, Mcceldon JL : The basal cell nevus syndrome. J Am Dent Assoc, 190:274-277, 1964. https://doi.org/10.1001/jama.1964.03070170015003
  10. Myoung H, Hong SP, Hong SD, et al. : Odontogenic keratocyst : Review of 256 cases for recurrence and clinicopathologic parameters. Oral Surg Oral Med Oral Pathol, 91:328-333, 2001. https://doi.org/10.1067/moe.2001.113109
  11. Diaz-Fernandez JM, Infante-Cossio P, Belmonte- Caro R, et al. : Basal cell nevus syndrome. Presentation of six cases and literature review. Med Oral Patol Oral Cir Bucal, 10:E57-66, 2005.
  12. Phillipsen H : On keratocysts in the jaws. Tandleagebladet, 60:963, 1956.
  13. Madras J, Lapointe H : Keratocystic odontogenic tumour : reclassification of the odontogenic keratocyst from cyst to tumor. J Can Dent Assoc, 74:165-165h, 2008.
  14. Wright JM : The odontogenic keratocyst : orthokeratinized variant. Oral Surg Oral Med Oral Pathol, 51:609-618, 1981. https://doi.org/10.1016/S0030-4220(81)80011-4
  15. Toller P : Origin and growth of cysts of the jaws. Ann R Coll Surg Engl, 40:306-336, 1967.
  16. Voorsmit RA, Stoelinga PJ, van Haelst UJ, et al. : The management of keratocysts. J Maxillofac Surg, 9:228-236, 1981. https://doi.org/10.1016/S0301-0503(81)80049-5
  17. Scharffetter K, Balz-Herrmann C, Lagrange W, et al. : Proliferation kinetics-study of the growth of keratocysts. Morpho-functional explanation for recurrences. J Craniomaxillofac Surg, 17:226-3, 1989. https://doi.org/10.1016/S1010-5182(89)80074-5
  18. Soskolne WA, Shear M : Observations on the pathogenesis of primordial cysts. Br Dent J, 3:123:321-326, 1967.
  19. Mustaciuolo VW, Brahney CP, Aria A : Recurrent keratocysts in basal cell nevus syndrome : review of the literature and report of a case. J Oral Maxillofac Surg, 47:870-873, 1989. https://doi.org/10.1016/S0278-2391(89)80051-5
  20. Kolokythas A, Fernandes RP, Pazoki A et al. : Odontogenic keratocyst : to decompress or not to decompress? A comparative study of decompression and enucleation versus resection/peripheral ostectomy. J Oral Maxillofac Surg, 65:640-644, 2007. https://doi.org/10.1016/j.joms.2006.06.284
  21. Ahlfors E, Larsson A, Sjogren S : The odontogenic keratocyst : a benign cystic tumor? J Oral Maxillofac Surg, 42:10-19, 1984. https://doi.org/10.1016/0278-2391(84)90390-2
  22. Pogrel MA, Jordan RC : Marsupialization as a definitive treatment for the odontogenic keratocyst. J Oral Maxillofac Surg, 62:651-655, 2004. https://doi.org/10.1016/j.joms.2003.08.029
  23. Myoung H, Hong SP, Hong SD, et al. : Odontogenic keratocyst : Review of 256 cases for recurrence and clinicopathologic parameters. Oral Surg Oral Med Oral Pathol, 91:328-333, 2001. https://doi.org/10.1067/moe.2001.113109
  24. Muzio L, Nocini P, Bucci P, et al. : Early diagnosis of nevoid basal cell carcinoma syndrome. J Am Dent Assoc, 130:669-674, 1999.