Case Report

Nutritional rickets in a 2-year-old child: case reportDonal McDonnell, BDS, FFD, MSc, FRCD(C) Gary Derkson, DMDLewei Zhang, BDS, PhD, FRCD(C) Jean Hlady, MD, FRCP(C)

Osteomalacia is a disorder of the adult skeletonin which mineralization of newly formed boneis defective. In children, the disease is mani-

fested as rickets. Bone mineralization requires normalplasma concentrations of calcium, phosphate, and vi-tamin D. A deficiency of these factors results in im-paired cartilage calcification, widening of the ends oflong bones, abnormal mineralization of bone matrix,and skeletal deformities.

Vitamin D is a fat-soluble vitamin that acts verymuch like a hormone in the control of calcium andphosphorus homeostasis. In general, vitamin D is re-sponsible for calcium uptake from the intestine, bonecalcium mobilization, and renal calcium reabsorption.In vitamin D deficiency, the accumulation of osteoidand the lack of mineralization result in a soft skeleton,deformity, or fracture.

Rickets usually is classified according to the under-lying cause. Hutchison and Bell1 indicate that the mostcommon cause of vitamin D deficiency in the UnitedStates is intestinal malabsorption associated with dis-eases of the small intestine, hepatobiliary tree, and pan-creas. Nutritional deficiency and sunlight deprivationare uncommon causes, and with the addition of vita-min D as a food additive, nutritional rickets is now rarein North America.1-2 However osteomalacia has beenreported in immigrant populations in the United King-dom and is attributed to ethnic traditions such as tra-ditional clothing, women staying indoors, and dietarypatterns.3'5

This report illustrates a case of nutritional rickets ina 2.5-year-old child born in Canada who had dentalradiographic evidence of rickets affecting the jaws. Themedical, hematological, and radiological investiga-tions, and histopathological findings of the extractedteeth are reported. A successful response to treatmentis illustrated with changes in the radiological appear-ances of the jaws.

Case reportChief complaint and history of present illness

A female, aged 2 years and 6 months, was referredby her general dentist to British Columbia's Children'sHospital dental department with facial pain and swell-ing. Oral antibiotics had been prescribed and the symp-

toms were improving when the child was first seen.The child was born in Canada to non-English speak-ing Vietnamese parents. Clinical examination was dif-ficult due to lack of cooperation, but did reveal manygrossly carious teeth. Arrangements were made forexamination and treatment under general anesthesia.

Physical examinationThe patient weighed 19.2 kg (> 95th percentile) and

was 87 cm tall (25th percentile). Preoperative assess-ment by the pediatrician revealed a waddling gait, anda hip radiograph (not shown) showed normal acetabu-lar angles, metaphyseal lucency, and medial beaking.Hemoglobin was within normal limits.

Oral examinationUnder general anesthesia in the operating room,

clinical examination confirmed a complete primarydentition and a discharging sinus in the buccal sulcusassociated with the carious maxillary right primary firstmolar. Carious lesions were noted in the followingteeth: all maxillary primary incisors, all primary firstmolars, and the mandibular primary central incisors.

Fig 1. Intraoral radiographs showing the grossly cariousteeth, the size of the pulp chambers, the crypts of the de-veloping teeth, the outline of which is attenuated (as isthe lamina dura). The trabecular pattern is sparse, andthe inferior cortex on the left side appears thinned. Theendotracheal tube accounts for the radiopacity in the in-cisor region.

Pediatric Dentistry - 19:2,1997 American Academy of Pediatric Dentistry 127

TABLE. NORMAL, BASELINE AND POST-TREATMENT CALCIUM,

PHOSPHORUS AND ALKALINE (Alk) PHOSPHATASE

Fig 2. Hand radiographs showing widening of thegrowth plates and generalized demineralization.

Intraoral dental radiographs (Fig 1) illustrate the extentof the carious lesions and the large size of the pulpchambers with pulpal horn extension close to thedentoenamel junction, especially in the mandibularprimary first molars. Hypoplasia was not evident in theunerupted teeth. The bone trabeculation in the anteriormandible and maxilla was very sparse. The laminadura and the crypts of the developing teeth werepresent, but attenuated. On the left side, the cortices ofthe inferior alveolar canal were evident and the infe-rior cortex of the mandible appeared thinned, but theother side was not available for comparison.

Dental treatment

The maxillary primary central and lateral incisorteeth and the maxillary primary first molar teeth wereextracted and submitted for histopathological exami-nation. Hemorrhage was controlled easily by local pres-sure and postoperative recovery was uneventful.

Supplemental testing

Further hematological and radiological investiga-tions were carried out. The alkaline phosphatase levelwas 2264 U/L, approximately seven times the upperlevel of the normal range, and both serum calcium andphosphorus levels were reduced (Table 1). Hand (Fig2) and knee radiographs (not shown) showed sparsetrabeculation with generalized demineralization and alack of cortical outline. Some growth plates were wid-ened and showed findings consistent with rickets. Di-etary analysis revealed a diet of rice and various typesof Vietnamese foods that were low in calcium but highin calories. About 1 L of soya milk (not vitamin D-for-tified) was consumed per day, with no cow's milk.

Histological examinationWhen extracted teeth were examined microscopi-

cally, all were found to be severely carious with a thicklayer of dental plaque directly abutting the cariousdentin. The dentinal tubules seemed unremarkable inthe crowns but decreased markedly in the roots. The

Calciummmol/L

Phosphorusmmol/L

Alk PhosphataseU/L

NormalBaselineCompletion

2.17-2.452.142.39

1.32-2.100.791.56

145-3202264

26

width of the predentin layer in the crowns seemed nor-mal, but was three to four times wider than normal inthe molar roots and two to three times wider than nor-mal in the incisor roots (Fig 3). Irregularity of the den-tin-predentin border, interglobular dentin, and focalareas of external root resorption were noted in all teeth.In areas with no root resorption, the thickness of ce-mentum varied from normal to hardly identifiable.Bone trabeculae present were primarily immature wo-ven bone and showed widened osteoid seams.

Medical managementThe clinical, biochemical, and radiographic evidence

confirmed the diagnosis of rickets. Oral vitamin D andCalcium Sandoz (Sandoz Canada Inc., Dorval, QB)were prescribed. An initial drop in the serum calciumlevel was easily corrected by adjusting the medication.The child was discharged after 10 days and reviewedon an outpatient basis. With treatment and a change indiet to include cow's milk, the alkaline phosphatase,

Fig 3. Photomicrographs showing markedly widened pre-dentin (long arrows) in the presence of interglobular den-tin (short arrows) in the root of an incisor tooth.

calcium, and phosphorous returned to normal levels,thus confirming a nutritional cause of the rickets andexcluding other causes. Follow-up radiographs of thehands (Fig 4) and knees (not shown), showed evidenceof healing. Walking had improved and the waddlinggait had disappeared. The child's weight was stillabove the 95th percentile and her height had increasedto the 80th percentile. After 8 months, the patient wasdischarged to the care of her family physician.

The patient was lost to dental follow-up for 2 years.On further review, the remaining carious teeth were

128 American Academy o/Pediatric Dentistry Pediatric Dentistry-39:2, 2997

Fig 4. Post-treatment radiographs showing improvementin the appearance of the growth plates, which are moresharply defined and better mineralized than in Fig 2.

Fig 5. Intraoral radiographs dated 2 years after beginning treat-ment, illustrating better outlining of the lamina dura, the inferiorcortex of the mandible, and increase in the trabecular patternwithin the mandible. All permanent second premolars suggest hy-poplasia of the crowns. The endotrachael tube is visible in the up-per incisor region.

restored and radiographs showed a normal trabecularpattern, an improvement in the outline of the cryptcortices around the unerupted teeth, and a denser in-ferior cortex of the mandible (Fig 5). This pattern ofimprovement was similar to the changes seen in theother bones.

Discussion

Nutritional rickets is rare in North America, and thisreport illustrates the investigation and medical man-agement of a patient who initially presented with fa-cial cellulitis coincidental to the diagnosis of rickets.The diagnosis of rickets based on dental radiographicchanges alone may be difficult as there may be no spe-cific features evident.6"8 Poyton and Pharoah7 report thefollowing radiographic features may be seen: os-teoporosis, the crypts of the unerupted teeth andlamina dura may be attenuated, and hypoplasia ofenamel may occur. Goaz and White1* and Gibilisco10 re-port thinning of the inferior cortex of the mandible and

state that radiographic defects in the jaws in nutritionalrickets are uncommon.

The large pulp chambers and pulp horns, at the ex-pense of dentin, combined with the extent of dentalcaries present are perhaps more suggestive of vitaminD resistant rickets.6-"•12 Osteoporosis and hypo-phosphatasia may give a similar appearance, whichcannot be differentiated from rickets on the radio-graphic features alone.

Some studies indicate that clinical evidence ofenamel hypoplasia is found in a high percentage ofrachitic children,13-u although others found no corre-lation between enamel hypoplasia and rickets.15 Opin-ion is divided on the susceptibility of hypoplastic teethto caries.16"18 However, there is general agreement thatonce caries has started in the hypoplastic tooth, itspreads more rapidly than in a nonhypoplastic tooth.Vitamin D deficient children have been found to haveretarded eruption.19 Few studies have reported the his-

topathological changes in dentin or cementum ofvitamin D deficient patients. Recent studies arerare due to the rarity of rickets.

The general goals of medical treatment of rick-ets have been well described by Hutchison andBell.1 Vitamin D is the drug of choice and calciumsupplements are also frequently used. Calciumabsorption is usually low in patients with rick-ets; increasing calcium intake reduces the dose ofvitamin D related compounds required for treat-ment. Diet analysis and investigation to excludecauses other than nutrition should be under-taken. Treatment continues until there is radio-graphic evidence of healing and a return to nor-mal plasma concentrations of calcium,phosphorus, and alkaline phosphatase. The long-term prognosis should be good.

Nutritional rickets is a rare occurrence inNorth America, but with changing patterns of immi-gration and dietary habits associated with such popu-lations, its occurrence may increase. The radiographicfeatures suggestive of rickets in this patient at presen-tation showed changes in appearance following treat-ment, namely improvement in crypt outline, increasein trabeculation, and increase in density of the man-dibular cortex. This case is reported because it illus-trates some radiographic evidence of rickets in the jawbones, which changed in appearance in response to ap-propriate medical treatment.

Dr. McDonnell is assistant professor and Dr. Zhang is associateprofessor, Department of Oral Medical and Surgical Sciences, Uni-versity of British Columbia, Vancouver. Dr. Derkson is associateprofessor and head, Department of Dentistry and Dr. Hlady is aclinical professor, Department of Pediatrics, both at BritishColumbia's Children's Hospital, Vancouver. Mr. Harold Traegerphotographed the illustrations.

1. Hutchison FN, Bell NH: Osteomalacia and rickets. SeminNephrol 12:127-45,1992.

2. Manologas SC, Olefsky JM: Metabolic Bone and Mineral Dis-orders. New York: Churchill Livingstone, 1988, pp 103-92.

Pediatric Dentistry - 19:2,1997 American Academy of Pediatric Dentistry 129

3. Stamp TCB, Exton-Smith AN, Richens A: Classical ricketsand osteomalacia in Britain. Lancet 2:308, 1976. [Letter]

4. Holmes AM, Enoch BA, Taylor JL, Jones ME: Occult rick-ets and osteomalacia amongst the Asian immigrant popu-lation. Q J Med 42:125-49, 1973.

5. Pietrek J, Preece MA, Windo J, O’Riordan JLH, DunniganMG, McIntosh WB, Ford JA: Prevention of Vitamin D defi-ciency in Asians. Lancet 1:1145-48, 1976.

6. Worth H: Principles and Practice of Radiographic Interpre-tation. Chicago: CV Mosby Co, 1963, pp 341-43.

7. Poyton HG, Pharoah MJ: Oral Radiology, 2nd ed. Philadel-phia: BC Decker Inc. 1989, 258-61.

8. Miles DA, Van Dis M, Kaugars GE, Lovas JGL: Oral andMaxillofacial Radiology: Radiologic/Pathologic Correla-tions. Philadelphia: WB Saunders Co. 1991, p 244.

9. Goaz PW, White SC: Oral Radiology: Principles and Inter-pretation, 2nd ed. St Louis: CV Mosby Co. 1987, pp 643-45.

10. Gibilisco JA, Ed. Stafne’s Oral Radiographic Diagnosis, 5thed. WB Saunders Co. 1985, pp 277-78.

11. Farman AG, Nortj6 CJ, Wood RE: Oral and Maxillofacial Di-agnostic Imaging. St Louis: Mosby-Year Book Inc. 1993, pp338-42.

12. Langlais RP, Langland OE, Nortj6 CJ: Diagnostic Imaging ofthe Jaws. Baltimore: Williams & Wilkins. 1995, pp 454-57

13. Dick JL: The teeth in rickets. Proc R Soc Med 9:83-91, 1916.14. Grahn6n H, Selander P: The effect of rickets and spasmo-

philia on the permanent dentition. Odontol Revy 5:7-26,1954.

15. Marshall-Day CD: Nutritional deficiencies and dental car-ies in Northern India. Brit Dent J 76:115-22, 143-47, 1944.

16. Anderson PG, Williams CHM, Halderson H, SummerfeldtC, Agnew RS: The influence of vitamin D in the preventionof dental caries. Am Den Ass J 21:1349-66, 1934.

17. Bibby BG: The relationship between microscopic hypopla-sia and dental caries. J Dent Res 22:218, 1943. [Abstr #60]

18. Shelling DH, Anderson GM: Relation of rickets and vitaminD to the incidence of dental caries, enamel hypoplasia andmalocclusion in children. Am Den Assoc J 23:840-46,1936.

19. Speidel TD, Stearns G: Relation of vitamin D intake to ageof infant at time of eruption of first deciduous incisor. JPediatr 17:506-11, 1940

The Newest Revelation from Kinder Krowns

Incisa

incisaLock is an Exclusive Feature From Kinder Krowns

¯Save Operatory Time¯Get Consistent Results

Aesthetic Aitemative"

Ask About our Krown Shortening Service[If yo~ find that the stainless steel krowns are too long.

right out of the box, we can trim them for you before weput the facing on; saving you valuable time.

The new Kinder Krowns combine the bestof the original with some revolutionary newideas in pediatric restorations. A new hybridcomposite which has a natural translucencymimics childrens teeth even better than before.

The most exciting concept in the newkrowns is what we call IncisaLock. We usemechanical retention at the incisal area, theweakest point of ANY of these types ofkrowns. Our findings show in the majority ofcases the incisal portion of the krown is filledwith cement. We have utilized that to ouradvantage. The internal composite also helpsstrengthen the bond of the krown to the tooth.

Kinder Krowns are available in two shades,Pedo 1 and Pedo 2, and we specialize inkustomizing the krowns to your individualspecifications. But perhaps the best reason touse Kinder Krowns is that they look like teeth.We don’t do Chiclets! Call today!

MAYCLIN DENTAL STUDIOS, INC.2629 LOUISIANA AV~. SOUTH " M~rq~.APOL~S, MN 55426

1-800-522-7883(612) 926-1809, FAX (612) 926-6331

~,ALL OR F~X TOD~Y FOR ~OR$ INFO OR TO TI~Y OUR RI$1&FI~cE II~TRO

130 American Academy of Pediatric Dentistry Pediatric Dentistry - 19:2, 1997