Micrognathia after radiation therapy for childhood facial tumors

Micrognathia after radiation therapy for childhood facial tumors

Micrognathia after radiation therapy for childhood facial tumors Report of two cases with long-term follow-up Sue C. Kaste, DO,a and Kenneth P. Hopkin...

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Micrognathia after radiation therapy for childhood facial tumors Report of two cases with long-term follow-up Sue C. Kaste, DO,a and Kenneth P. Hopkins, DEPARTMENTS HOSPITAL

OF DIAGNOSTIC AND

As the survival

UNIVERSITY duration

and cure

IMAGING

AND

DDS, MSb Memphis, DENTISTRY,

ST. JUDE

Tenn.

CHILDREN’S

RESEARCH

OF TENNESSEE rate of children

with

malignant

conditions

improve,

awareness

of long-term

effects is crucial. Two case reports demonstrate long-term changes in dentition and orofacial development in children ‘who been treated for facial malignant conditions. Changes included mandibular and maxillary hypoplasia, partial anodontia, microdontia, altered eruption pattern, root stunting, and altered ametogenesis. (ORAL SURG ORAL MED ORAL PATHOL 1994;n:95-9)

Inherent in the improved survival of children treated for childhood malignant conditions are potential adverse long-term side effects of radiotherapy and chemotherapy. Radiation-induced effects include maxillary and mandibular hypoplasia, tooth or root dwarfism, anodontia, caries, and altered tooth eruption patterns.‘-** Most children receive more than one type of therapy, often making it difficult to distinguish the sequelae of chemotherapy from radiation effects.1-3, 23 The impact and severity of potential side effects vary with the child’s age at the time of therapy, the radiation dose delivered, atid the anatomic location treated.13p 14,I’, 24 Radiation effects on facial bones result from osteocyte death, microvascular injury, periosteal damage, and fibrous replacement of marrow spaces. These changes lead to altered bone growth and development.7y *, ‘** 16y“3 *’ This article reports the long-term follow-up of two children treated with radiation and chemotherapy for facial malignant conditions. CASE 1

A 4-year-oldblack malepresentedwith a rapidly enlarging fixed, ulceratingsoft tissuemassof the palate.The mass involved the left soft palate, left nasopharynx, and left parapharyngealarea. Computedtomography showedthe lesionto extend from the baseof the skull to C2 and from the pterygoid platesposteriorlyto the vertical ramusof the mandible,crossingbetweenthe zygomaand the lateral wall of the maxillary antrum. There wasdestructionof the lat-

Supported in partby theNationalCancerInstituteCancerCenter (CORE)grantCA 21765andby theAmericanLebanese SyrianAssociated Charities. aDepartment of Diagnostic Imaging. bDepartment of Dentistry. Copyright@1994by Mosby-YearBook,Inc. 0030-4220/93/$1.00 + .lO 7116151346 Support

side had

era1pterygoid plate. Biopsy revealedan extra-abdominal desmoidtumor (aggressivefibromatosis). The lesionwasinoperable.Treatmentwith dactinomycin initially resultedin mild regressionof the lesion.After three coursesof chemotherapy,however,the tumor progressed. The patient wasthen given 32 Gy therapeuticmidplaneirradiation in 21 fractions over 30 days, using parallelopposedlateral fields(Fig. 1, A). The field borderswerethe top of sellato the bottom of mandiblecraniocaudallyand posteriormandibleto posterioronethird of nasalcavity anteroposteriorly.A lateral cephalometricradiographshowed a normalrelationshipbetweenthe mandibleand maxilla at the time of radiation treatment (Fig. 1, B). After radiation therapy, the tumor growth stabilizedfor 3 years. A panoramicradiographobtained2 yearslater (at age6) showeda lytic area in the left posteriormandiblewith displacementof the developingsecondpermanentmolar superiorly into the ramus.A panoramicradiographtakenat age 8 showed total root stunting of all first permanentmolars and lower incisorswith extensivelysisof the left mandibular ramus. Radiographicexamination at age 9 showed eruption of the maxillary central incisorswith normalroot formation andcompleteroot agenesis of all developingpermanent secondmolarsand bicuspids.There was further displacementof the lower left secondmolar into the ramus by a lytic lesion. A panoramicradiograph at age 12 showedtotal root stuntingof all mandibularpermanentteeth. There wasalso severeroot stunting of the maxillary first and secondpermanentmolars. Root formation of the maxillary incisors was normal. The maxillary primary molars were being over-retained.The developingmandibularleft secondpermanentmolarwasdisplacedhigh into the ramus.Mandibular micrognathiawasbecomingpronounced,with a severe overjet of the maxillary incisors. When the patient was seen at the age of 17 years, micrognathiawasapparent,and there wasmicrodontiaof all permanentsecondmolars(Fig. 1,C to E). Theunerupted and developinglower left secondand third molar crowns weredisplacedhigh into the left ramusalmostto the coronoid process.

95

%

Kaste and Hopkins

ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGY January 1994

Fig. 1. A, PhotographandB, corresponding lateral cephalometricradiographof d-year-oldpatientwho was treated with 32 Gy for fibromatosisof left soft palatewith involvementof nasopharynxand lateral pharyngeal wall.

CASE 2

A Syear-old white malewasdiagnosedwith embryonal rhabdomyosarcoma arising in the right nasopharynx.He wastreated with a four-drug chemotherapeuticregimenof cyclophosphamide, doxorubicin hydrochloride (Adriamytin), dactinomycin,andvincristine.Chemotherapywasfollowedby 35 Gy irradiation of the nasopharynxand right maxilla deliveredin 24 fractions via anterior field (Fig. 2, A and B). There wasno evidenceof diseaseat the conclusionof therapy. When the patient was 15 years old, severegeneralized root stuntingof the maxillary dentition and malpositionof the right maxillary canineincisor,permanentsecondbicuspid, and secondmolar wereradiographicallyevident (Fig. 2, C). There wasalsosignificantmaxillary hypoplasiaand facial disproportion(Fig. 2, D and E). Anodontia of the right maxillary and mandibularthird molarswasnoted. DISCUSSION

Animal models have demonstrated the marked radiosensitivity of developing teeth and facial bones. Doses of 10 Gy and 30 Gy permanently damaged mature ameloblasts, arresting tooth development. Case reports in the literature suggest that radiation

doses in excess of 20 Gy result in significant bone growth disturbance regardless of the child’s age, whereaslower dosesin children 1 to 2 years old result in minor or no residual deformity.t9 Insults occurring during periods of maximum bone growth and development, that is, birth to 6 years of age and during puberty, result in particularly marked growth disturbance.” Radiation damage to bone, periodontal ligament, and pulp occurs simultaneously. The effects on these structures are interrelated, and it is difficult to distinguish between primary and secondary effects.19 Our two patients received 32 and 35 Gy facial irradiation at early ages. The subsequentmicrodontia and root stunting may have been as a result of chemotherapy, radiation therapy, or their combined effects. However, the patterns of these abnormalities were commensurate with radiation damage at an early age. In patient 1, marked root stunting of all primary teeth was expected becausethe crowns had already formed at the time of therapy although the roots were still developing. Microdontia of the left maxillary bicuspid paralleled its developmental lag behind that of the right, as observed on the initial

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Fig. 1. cont. C, PhotographandD, corresponding lateral cephalometricradiographof samepatient 13years later showmarkedmandibularhypoplasia.E, Panoramicradiographshowsroot stuntingof all permanent teeth, microdontiaof maxillary secondpermanentmolars(arrows), and malpositioningof mandibularteeth (arrowhead) in rami.

panoramic radiograph. Maxillary third molar microdontia could be expected becausethe tooth buds had not formed at the time of therapy. The severedisturbance of mandibular growth and the relative sparing of the maxilla reflect the distribution of the radiation field. The dental abnormalities in patient 2 were similar to those in patient 1 but lesssevere. In this patient, the less severe impairment of mandibular dentition and bony development correlates with radiation therapy directed to the maxilla and nasopharynx.

CONCLUSION We have followed two patients who received multimodality therapy for facial malignant conditions at an early age. Their facial developmental abnormalities occurred at the sites of local irradiation, which suggeststhe causal involvement of radiation therapy. With the improved cure rates for childhood malignant conditions in the past decade, late effects of therapy must be recognized to minimize their impact on the quality of life in long-term survivors. Identification and treatment of these sequelaemay prove to

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ORAL

Fig. 2. A, Photograph and B, corresponding lateral diagnosed rhabdomyosarcoma of the nasopharynx maxillomandibular relationship.

be as challenging as management lignant condition.

of the primary ma-

We thank tion.

manuscript

Ms.

Patsy

Burnside

for

prepara-

REFERENCES 1. Dahllof G, Barr M, Bolme P, et al. Disturbances in dental development after total body irradiation in bone marrow transplant recipients. ORAL SURG ORAL MED ORAL PATHOL 1988;65:41-4. 2. Jaffe N, Toth BB, Hoar RE, Reid HL, Sullivan MP, McNeese MD. Dental and maxillofacial abnormalities in long-term survivors of childhood cancer: effects of treatment with chemotherapy and radiation to the head and neck. Pediatrics 1984;73:816-23. 3. Berkowitz RJ, Neuman P, Spalding P, Novak L, Strandjord MD, Coccia PF. Developmental orofacial deficits associated with multimodal cancer therapy: case report. Pediatr Dent 1989;11:227-31. 4. Le Masney NJ, Holland T, O’Brien C, O’Mullane DM. The effects of antimalignancy therapy as seen in the oral cavity of childrenandyoungadults: a review. J Ir Dent Assoc 1990;36:4752. 5. Maguire A, Craft AW, Evans RGB, et al. The long-term effects of treatment on the dental condition of children surviving malignant disease. Cancer 1987;60:2570-5. 6. Poyton HG. The effects of radiation on teeth. ORAL SURG ORAL MED ORAL PATHOL 1968;26:639-46. 7. Byrne JJ. The effect of ionizing irradiation on the developing maxilla and mandible. Am J Orthodontics 1961;47:646-60.

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SURGERY

ORAL

MEDICINE

ORAL

PATHC Januar

Ti? 53 ‘I’ I‘YY4

radiograph of a j-year-old hoy with newly dental development pattern and normal

8. Rohrer MD, Kim Y, Fayos JV. The effect of cobalt-60 irradiation on monkey mandibles, ORAI SLRG ORAL MED ORAL PATHOL 1979;48:424-40. 9. Anneroth G, Holm L-E, Karlsson G. ‘The effect of radiation on teeth: a clinical, histologic, and microradiographic study. Int J Oral Surg 1985;14:269-74. 10. McGinnis JP, Hopkins KP. Thompson EI, Hustu HO. Mandibular third molar development after mantle radiation in long-term survivors of childhood Hodgkin’s disease. ORAI. SURG ORAL. MEL) ORAL PATHOL 1987;63:630-3. 11. Pietrowski J. Menczel J. Tooth dwarfism and root underdevelopment following irradiation. ORAL SHIRT ORAL MED ORAL PATHOL 1966;22:95-9. 12. Doline S, Needleman HL, Petersen RA, Cassady JR. The effect of radiotherapy in the treatment of retinoblastoma upon the developing dentition. J Pediatr Ophthalmol Strabismus 1980;17:109-13. 13. Takeda Y. Kuroda M, Amari E, Yanagisarva T. Failureof root development of human permanent teeth following irradiation. Int J Oral Maxillofac Surg 1987;16:376-82. 14. Guggenheimer J, Fischer WG, Pechersky JL. Anticipation of dental anomalies induced by radiation therapy. Radiology 1975;117:405-6. 15. Gowgiel JM. Eruption of irradiation-produced rootless teeth in monkeys. J Dent Res 1961;40:538-47. 16. Savostin-Asling K, Silverman S. Effects of therapeutic radiation on microstructure of the human mandible. Am J Anat 1978;151:295-306. 17. Nwoku AL, Koch H. Effect of radiation injury on the growing face. J Maxillofac Surg 1975;3:28-34. 18. Guyuron B, Dagys AP, Munro IR, Ross RB. Effect of irradiation on facial growth: a 7- to 25-year follow-up. Ann Plast Surg 1983;11:423-7.

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Fig. 2. cont. C, Panoramic radiograph 10 years after radiation therapy to 35 Gy shows severe root of the maxillary dentition and malpositioning of the right maxillary canine incisor, second bicuspid, ond molar. There is also anodontia of the right maxillary and mandibular third molars. Note relative of the mandibular dentition. D, Corresponding photograph and E, lateral cephalometric radiograph sultant maxillary hypoplasia and root stunting.

19. Carpenter JS. Dental care for children who have received head and neck therapeutic radiation. J Pedod 1978;3:36-5 1. 20. Nathanson A, Backstrom A. Effects of 60CO-y-Irradiation on teeth and jaw bone in the rabbit. Stand J Plast Reconstr Surg 1978;12:1-17. 2 1. McGinnis JP, Hopkins KP, Thompson EI, Hustu HO. Tooth root growth impairment after mantle radiation in long-term survivors of Hodgkin’s disease. J Am Dent Assoc 1985; (111):584-S. 22. Hazra TA, Shipman B. Dental problems in pediatric patients with head and neck tumors undergoing multiple modality therapy. Med Pediatr Oncol 1982;10:91-5.

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23. Rosenberg SW. Chronic dental complications. Monogr Nat1 Cancer Inst 1990;9:173-8. 24. Eckles TA, Kalkwarf KL. Impaired tooth root development after treatment of a cerebellar astrocytoma: a case report. ORAL SURG ORAL Reprint

MED ORAL PATHOL

requests:

Sue C. Kaste, DO Department of Diagnostic Imaging St. Jude Children’s Research Hospital 332 N. Lauderdale Memphis, TN 38105

1989;68:414-8.