200 Australian Dental Journal, August, I960

Dental sequelae in deciduous dentition in vitamin D resistant rickets

Case Report

Robert Harris* and

Rickets is a disease of childhood in which the important changes are those occurring in the bones, calcification being delayed. Eruption of the teeth is retarded, and Mead(” is of the opinion that they are often deformed and very susceptible to caries, crowding of the arches being common. Stones(*) suggests that there may be some doubt about this and other authors support him. In the Punjab a hiqh prevalence of rickets associated with a deficiency of calcium, phosphorus and vitamin D in the diet was associated with little hypo- plasia and/or dental caries (Taylor and Day‘3’). Weinmann and Schour‘‘) claim that enamel formation and calcification :ire apparently unaffected by vitamin D deficiency, unless it is severe and associated with other Pactors; however, the dentine is alwa)s involved and interglobular dentine is observed. Staz‘’) examined 61 Indian children who were vegetarians and all had hypoplasia, but only three had rickets, whereas 37 suffered sljme exanthematous disease (two of those with rickets were in this group). Sjoquist‘‘) also concluded that some other factor (spasmo-

* Assistant Superintendent and Head, Depart- ment of Preventive Dentistry, l’nited Dental Hos- pital, Sydney.

t Assistant Pirector, Institute of Dental Ilesearch, United Dental Hospital, Sydney. ( I ) Mead, S. \’.-Oral Surgery. St. Louis : C. V.

Mosby, 4th ed., 1!1.54 (p. 8 6 8 ) . (*) Stones, H. Fi.--Oral and Dental Disenses.

Edinburgh: $2. and S. Livingstone, 3rd c.3., 1 9 5 4 (p. 7 6 ) .

(:I) Taylor, G, F., ancT Day, D. I). &I.-Relation of vitamin D and mineral deficiencies to dental caries. Brit. JI. .J., May F, D. 919, 1939.

(I) Weinmann. J. P., and Schour. I.--F:xperimentnl studies in calrifiwtion. 1. Effect of rachito- genic diet on dental tissues of white ra t . Am.

(m Staz, J.-Hypoplasic teeth and dental caries. C.A. Med. J.. I i : 1 ( J a n . ! i ) 1 9 4 3 ,

c6) Sjoquist, P. IT.-The macroscopic formation of the enamel deformation caused by rickets ur spasmophilia and a theory of their mechanirm of evolution. Acta P e d . , 1 9 : 281, 1937 .

J. I’dth., 2 1 : 821 . 1 9 4 5 .

Harold R. Sullivan?

philia) was necessary f o r disturbance of enamel formation. Moore”) states that rickets does not appear for some weeks or months after birth and congenital rickets has neyer been observed. Follis,(R) discussing the possible etiology of vitamin D resistant rickets, said that “an increasing number of isolated reiisl tubular defects have come to be recognized. These are associated with a n inability to resorb certain metabolites. . . . Certain of these defects may lead to rickets or osteomalasia. The mcst important is a syndrome which usually has an hereditary background and makes its appear- ance in childhood or in early adulthood.”

It can be considered, therefore, that rickets of itself does not cause disturbances in forma- tion of the enamel, but the dentine formation is deficient and there are well-defined changes in bone.

Case Report

A boy, aged seven years and three montns, attended for examination and treatment en 9.9.59. He had been referred by his dentist following the development of a series of recnr- rent periapical infections on deciduous terth w-hich clinically were caries free, but the upper deciduous centrals were discoloured.

Previous his tory He had been exainined by his dentist dn

3.2.59, when the only oral abnormality observ2d was discoloration of the upper centrals. He was then seen on 31.3.59. when he had acute periapical infection on the deciduous lowc-r first molars and sinuses cn the labial gingivae of the upper central and lateral incisors. Vie acute conditior,s commenced to subside within 24 hours, bJt there was evidence that a n acute infection was affecting the deciduous upper left first mclar. Hematological examination on 2.4.59 showed little abnormality other than 16 per cent eosinophils. On 11.4.59 the acute infection appeared to hav3 completely subsided

(7) Moore, R. -4.-Textbook of Pathology. Phi!a-

CL) Follis, K. H.-Deficiency Disease. SpringAe!d : delphia: W. B. Saunders, 1 9 4 4 (p. i 2 C ) .

Charles C. Thomas, 1958 (p. 368).

Australian Dental Journal, August, I960 20 I

Fig. 1.-Sinuses can be seen above the four upper incisor teeth. The discolouration of the centrals is not so apparent in the photograph.

and the deciduous lower left central and the lower right and left lateral incisors were extracted. An examination on 11.7.59 revealed little evidence of the previous condition, but on 8.9.59 a n exacerbation occurred with other teeth becomina involved.

Examination 1. The boy appeared to have normal trunk

and upper limbs with some slight bossing of the forehead, but he showed marked bowing of the femora and tibia which had bern present since. he was two years old. His mother had obvious bow legs which had cot been improved by surgery. Another child showed no abnormality.

2. The child’s mouth had been well cared for, but both upper central incisors were dis- coloured. The permanent lower central incisors had erupted, and these with the eighteen deciduous teeth were caries-free, but there was some occlusal wear. The gingival margins

Fig. $.--Swelling caused by chronic abscess a t the apex of the lower left canine.

were firm and no signs of gingivitis could be seen.

Inflamed areas were present in the labial sulcus at about the apical depth of the upper anterior teeth, the upper left and lower left canines and the lower right first molar. Sinuses were present, but there appeared to be no dis- charge of pus (Figs. 1 and 2 ) .

3. X-ray examination. 9.9.59. Periapical films disclosed little evidence of resorption of the roots of the deciduous anterior teeth. Areas of rarefaction of alveolar bone could be seen at the apices of the lower left canines, lower right labial and lower first molars. Careful examination of the films also showed a n altera- tion in the opacity of the dentine of the second deciduous molars and of the unerupted first permanent molars. The lower central incisors also had some signs of abnormality. The roentgenograms confirmed the clini.?aI appraisal of freedom from caries. Lateral views of the mandible and skull revealed no osseous lesions.

Fig 3.-Periapical Alms showinrr differences in density in the radio-opacity of ihe dentine in both 1)ermanent and dccirluous teeth. Note also the abnorn~al size of the

pulp chambers in the u p p ~ r central incisors.

202 Australian Dental Journal, August , 1960

Fig, 4.-Very poorly calcified dentine seen in an upper deciduous incisor. This is typical of the dentine found in children suffering with

hereditarr vitamin D resistant rickets.

I*Yg. 6.--Ground section showing interglobular dentine throughout the entire depth of the dentine. Note also the scant but well-formed

enamel.

Fig. (i.-Groun:l s z t i o n showing malunion between the laliial and lingual dentine and loss of incisal enamel, permitting passage of organisms from the oral cavity into the exposed

Pulp.

4 . Laborutory investigations. ( a ) Haematological examination: No abnor-

( b ) Urinalysis: No abnormality. ( c ) Serological tests: Wassermann reaction,

negative; Kahn flocculation test, nega- tive.

Inorganic phosphate 2.3 mg. per 100 ml.

Alkaline phosphatase 35 King-Arnistroug

Calcium 10.0 mg. per 100 nil. serum.

Material obtained 17.9.59 from a wcll- developed abscess sac on the lower left canine. Smear: Numerous pus cells, occasional

Gram-positive cocci and fusiforni bacilli.

Culture: Mixed organisms, mainly alpha haemolytic streptococci; no recognizable pathogens.

Antibiotic sensitivity tests : Organisms sensitive to aureomycin, chloro- mycetin, penicillin, tetracycline, Gram-negative cocci resistant to erythromycin.

5. Treatment. As a prophylactic measure 600,000 I.U. of penicillin were given intra- muscularly on 7J0.59, 8.10.59 and 9.10.59. Under general anaesthesia the left upper central. lateral, canine, lower canine and first molar teeth, the right upper central, lateral, lowcr lateral, canine and first molar teeth were removed and material was obtained for examination. A slight rise in temperature persisted for two days, but healing was uneventful; the patient was discharged on 14.10.59.

6 . Pathological report. The material removed from the apical region of a n upper incisor proved to be par t of a n inflamed cyst. A series of decalcified sections prepared from a n upper incisor showed the pulp cavity and the dentine to be grossly infected, the pulp cavity containing purulent granulation tissue i n which were many cocci and rods.

The dentine was a classical example of inter- globular dentine (Figs. 4 and 5 ) . There was nialunion between the labial and lingual dentine. This was confirmed in a ground section (Fig. 6 ) where the thin incisal enamel was seen to have been worn away, allowing direct access from the oral cavity to the pulp chamber through the cleft between the lingual and labial dentine.

mality.

( r l ) Biochemical examination:

serum.

units per 100 ml. serum.

( e ) Bacteriological examination :

Discussion

Certain interesting features can be noted in

1. The child had bowing of the legs which

the history of this case.

was first noticed at two years of age.

Australian Dental Journal, August, I960 203

2. The mother had marked bowing of the legs and surgery was performed, but withcut marked improvement.

3 . The deciduous teeth were caries-free and the enamel was smooth.

4. Despite the freedom from caries, the pulps of various deciduous teeth progressively became infected and necrotic, resulting in 1)eriapical abscess formation which made their removal essential.

5. Resorption of the deciduous teeth and eruption of the permanent teeth were both delayed.

6. Laboratory investigation demonstrated a normal serum calcium, a lowered serum phos-

phorus and a n increased serum phosphatase relationship similar to that seen in rickets.

7. The pathological findings supported this and further demonstrated the possible path of infection to the pulp via the developmental defects i n the dentine. The amount of enamel covering the incisal portion of the teeth, as seen in the ground section, was slight, and occlusal wear apparently exposed the defective dentine to permit ready ingress of infection

United Dental Hospital. 2 Chalmers Street,

Sydney.

diistralian Art Our writers are beginning to write within the terms of this country, aud not of it. and our painters are beginning to do the same. The attitude of self-consciousness is snullling off, and the appearance of new forms will show themselves in a new and adult attitude. - Riisaell Drysdale, Lecture at the Art Gallery of New 8096th Wales, 1959.