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IJCRI 201 2;3(8):51 –56.<br />

www.ijcasereports<strong>and</strong>images.com<br />

Aswath et al. 51<br />

CASE IN IMAGES<br />

OPEN ACCESS<br />

Pimary hyperparathyroidism as central giant cell<br />

granuloma <strong>of</strong> the jaws: Pre­ <strong>and</strong> post­treatment pattern<br />

<strong>of</strong> clinical <strong>and</strong> radiographic presentation<br />

Nalini Aswath, Pravda Chidambaranathan<br />

ABSTRACT<br />

Introduction: The parathyroid gl<strong>and</strong>s regulate<br />

serum calcium <strong>and</strong> phosphorous levels by its<br />

secretion <strong>and</strong> maintenance, within physiological<br />

limits, <strong>of</strong> its hormone parathormone (PTH).<br />

Hyperparathyroidism is a metabolic disorder<br />

resulting from excessive secretion <strong>of</strong><br />

parathyroid hormone. Hyper secretion might<br />

occur either due to a primary pathology in the<br />

gl<strong>and</strong>s (or) due to secondary causes. Central<br />

giant cell lesions occur in jaw bones. <strong>Case</strong><br />

Report: A 20­year­old boy reported with<br />

complaints <strong>of</strong> swelling in left maxilla. Intra<br />

orally swelling extended from left upper canine<br />

till left upper second premolar <strong>and</strong> caused<br />

expansion <strong>of</strong> the cortical plates. Intra oral<br />

radiograph <strong>and</strong> orthopantomograph showed a<br />

poorly defined radiolucent lesion in between left<br />

upper canine <strong>and</strong> left upper 1st premolar with<br />

multiple cystic cavities in the region <strong>of</strong> the<br />

symphysis, left body <strong>and</strong> right angle <strong>of</strong><br />

m<strong>and</strong>ible. The radiographs <strong>of</strong> long bones showed<br />

osteoporosis. Serum alkaline phosphatase level<br />

was raised. Histopathology was reported as<br />

central giant cell granuloma. Nuclear scan <strong>of</strong><br />

parathyroid showed a functioning parathyroid<br />

adenoma. The swelling in left maxilla regressed<br />

Nalini Aswath 1 , Pravda Chidambaranathan 2<br />

Affiliations: 1<br />

Pr<strong>of</strong>essor & Head <strong>of</strong> Department-Sree Balaji<br />

Dental College <strong>and</strong> Hospital, Chennai, Tamilnadu, India;<br />

2<br />

Reader-Sathyabama Dental College <strong>and</strong> Hospital,<br />

Chennai Tamil Nadu, India.<br />

Corresponding Author: Dr. Nalini Aswath, “Mathuram”, Plot<br />

No:1 61 , #5, Murugu Nagar 5 th street, Velachery, Chennai,<br />

Tamil Nadu, India - 600042; Ph: +91 -044-22591 51 9; Mob:<br />

91 - 94441 -61 841 ; Fax: +91 -044-22460631 ; Email:<br />

naliniaswath@gmail.com<br />

Received: 20 December 2011<br />

Accepted: 1 2 April 201 2<br />

Published: 01 August 201 2<br />

on its own after the adenoma <strong>of</strong> the<br />

parathyroids was excised. Conclusion: Timely<br />

diagnosis <strong>of</strong> parathyroid adenoma results in<br />

total regression <strong>of</strong> intra oral swelling <strong>and</strong><br />

further progression <strong>of</strong> osteoporosis <strong>and</strong><br />

fractures <strong>of</strong> long bones can be prevented.<br />

Keywords: Central giant cell granuloma, Brown<br />

tumor, Hyperparathyroidism<br />

*********<br />

Aswath N, Chidambaranathan P. Pimary<br />

hyperparathyroidism as central giant cell granuloma <strong>of</strong><br />

the jaws: Pre <strong>and</strong> post treatment pattern <strong>of</strong> clinical <strong>and</strong><br />

radiographic presentation. <strong>International</strong> <strong>Journal</strong> <strong>of</strong><br />

<strong>Case</strong> <strong>Reports</strong> <strong>and</strong> <strong>Images</strong> 2012;3(8):51–56.<br />

*********<br />

doi:10.5348/ijcri­2012­08­167­CII­14<br />

INTRODUCTION<br />

The parathyroid gl<strong>and</strong>s regulate serum calcium <strong>and</strong><br />

phosphorous levels by secretion <strong>and</strong> maintenance,<br />

within physiological limits, <strong>of</strong> its hormone<br />

parathormone (PTH). Secretion <strong>of</strong> PTH is mainly<br />

controlled through interaction <strong>of</strong> calcium with calcium<br />

sensitive receptors on the membrane <strong>of</strong> the parathyroid<br />

cells. Hyperparathyroidism is a syndrome <strong>of</strong><br />

hypercalcemia resulting from excessive secretion <strong>of</strong><br />

parathyroid hormone. A case <strong>of</strong> central giant cell<br />

granuloma in left maxilla due to adenoma <strong>of</strong><br />

parathyroid gl<strong>and</strong> is presented.<br />

CASE REPORT<br />

A 20­year­old boy reported with complaints <strong>of</strong><br />

swelling in the left upper jaw (Figure 1). The swelling<br />

IJCRI – <strong>International</strong> <strong>Journal</strong> <strong>of</strong> <strong>Case</strong> <strong>Reports</strong> <strong>and</strong> <strong>Images</strong>, Vol. 3 No. 8, August 201 2. ISSN – [0976-31 98]


IJCRI 201 2;3(8):51 –56.<br />

www.ijcasereports<strong>and</strong>images.com<br />

Aswath et al. 52<br />

was initially smaller six months ago <strong>and</strong> gradually<br />

increased in size. It was asymptomatic since onset <strong>and</strong><br />

had not regressed in size at any time. There was no<br />

history <strong>of</strong> discharge from the swelling. No history <strong>of</strong><br />

paresthesia or fever. The past medical history <strong>and</strong><br />

surgical histories were unremarkable. Patient was<br />

moderately built, well nourished, calm <strong>and</strong> cooperative.<br />

His growth appeared to be retarded to his<br />

age. His vital signs were stable. A single left subm<strong>and</strong>ibular<br />

lymph node was enlarged, palpable,<br />

nontender, measuring 1 cm/1 cm in size, firm in<br />

consistency <strong>and</strong> mobile.<br />

Extra oral examination revealed a solitary swelling ,<br />

with well defined margins, measuring 3 cm/3 cm in<br />

diameter in the region <strong>of</strong> the left maxilla (Figure 2). It<br />

was irregular in shape, with a smooth surface extending<br />

anteriorly till nasolabial fold, posteriorly till the lateral<br />

margin <strong>of</strong> the eye, superiorly till the infra orbital<br />

margin, <strong>and</strong> inferiorly in line with angle <strong>of</strong> the mouth. It<br />

was nontender, hard in consistency. The skin over the<br />

swelling appeared normal. There was no local rise in<br />

temperature. The swelling was not mobile <strong>and</strong> it was<br />

fixed to the underlying structure.<br />

Intra oral examination (Figure 3) revealed a swelling<br />

extending from left upper central incisor to left upper<br />

second premolar. There was expansion <strong>of</strong> the buccal<br />

<strong>and</strong> palatal cortical plates. Margins were well defined. It<br />

was firm to hard in consistency, nontender. Mucosa<br />

over the swelling was normal. It measured 1.5 cm/2 cm<br />

in diameter. The left upper canine <strong>and</strong> first premolar<br />

were decayed <strong>and</strong> the vitality test showed a delayed<br />

response.<br />

Based on the history <strong>and</strong> clinical findings a provisional<br />

diagnosis <strong>of</strong> adenomatoid odontogenic tumor <strong>and</strong> a<br />

differential diagnosis <strong>of</strong> central giant cell granuloma,<br />

ameloblastoma, fibro­osseous lesion were given.<br />

Intra oral radiographs (Figure 4). Showed the<br />

presence <strong>of</strong> a poorly defined radiolucent lesion in<br />

between the left upper canine <strong>and</strong> left upper 1st<br />

premolar with divergence <strong>of</strong> roots <strong>of</strong> left upper 1st<br />

premolar. Orthopantomograph (Figure 5) showed the<br />

presence <strong>of</strong> a poorly defined radiolucent lesion in<br />

between the left upper canine <strong>and</strong> left upper 1st<br />

premolar with divergence <strong>of</strong> the roots <strong>of</strong> the left upper<br />

1st premolar <strong>and</strong> well defined cystic cavities in the<br />

region <strong>of</strong> symphysis, left body <strong>and</strong> right angle <strong>of</strong><br />

m<strong>and</strong>ible. Radiographs <strong>of</strong> long bones, chest, skull <strong>and</strong><br />

ultrasonography <strong>of</strong> abdomen were done. Radiographs <strong>of</strong><br />

long bones (Figure 6) were suggestive <strong>of</strong> osteoporosis.<br />

No significant findings were revealed in<br />

ultrasonography <strong>of</strong> abdomen.<br />

Biochemical investigations revealed the following:<br />

Serum calcium: 9.4 mg%, Serum phosphorus: 3.1 mg%,<br />

serum alkaline phosphatise: ­2559 mg/mL.<br />

Incisional biopsy was done <strong>and</strong> histopathological<br />

examination showed presence <strong>of</strong> mature bundles <strong>of</strong><br />

connective tissue with plump fibroblasts, interspersed<br />

with numerous multinucleated giant cells. Overlying<br />

epithelium appeared hyperplastic, with few bundles <strong>of</strong><br />

blood vessels. The lesion was suggestive <strong>of</strong> central giant<br />

cell granuloma (Figure 7).<br />

Thereafter the patient was referred to an<br />

endocrinologist to rule out hyperparathyroidism. After<br />

general examination <strong>of</strong> the patient by the<br />

endocrinologist, serum parathyroid estimation was<br />

done <strong>and</strong> was found to be elevated. Nuclear study <strong>of</strong> the<br />

parathyroid gl<strong>and</strong>s (Figure 8) was done with Tc­99m<br />

MIBI injected intravenously <strong>and</strong> it showed the presence<br />

<strong>of</strong> a functioning parathyroid lesion in the region <strong>of</strong> the<br />

lower pole <strong>of</strong> the left lobe <strong>of</strong> thyroid. An incisional<br />

biopsy was taken from the left. Inferior parathyroid<br />

gl<strong>and</strong> which was suggestive <strong>of</strong> parathyroid adenoma.<br />

The tumor was surgically excised <strong>and</strong> post­operatively<br />

the patient was treated with IV calcium <strong>and</strong> calcium<br />

supplements, <strong>and</strong> multivitamin tablets.<br />

The patient was periodically reviewed there­after at<br />

an interval <strong>of</strong> three months. The serum calcium,<br />

alkaline phosphatase, serum phosphorus returned to<br />

their normal limits.<br />

The central giant cell granuloma in the region <strong>of</strong> the<br />

left maxilla regressed on its own (Figure 9, 10).<br />

Intraoral radiographs, Occlusal X­ray (Figure 11),<br />

OPG (Figure 12), were taken which showed<br />

disappearance <strong>of</strong> the previous lesion. The multiple<br />

cystic cavities that were noticed in the m<strong>and</strong>ible too<br />

disappeared.<br />

Figure 1: Photograph <strong>of</strong> face shows swelling in the left maxilla.<br />

IJCRI – <strong>International</strong> <strong>Journal</strong> <strong>of</strong> <strong>Case</strong> <strong>Reports</strong> <strong>and</strong> <strong>Images</strong>, Vol. 3 No. 8, August 201 2. ISSN – [0976-31 98]


IJCRI 201 2;3(8):51 –56.<br />

www.ijcasereports<strong>and</strong>images.com<br />

Aswath et al. 53<br />

Figure 4: Intraoral radiograph shows poorly defined<br />

radiolucent lesion between canine <strong>and</strong> second premolar <strong>and</strong><br />

divergence <strong>of</strong> root <strong>of</strong> first premolar.<br />

Figure 2: Facial pr<strong>of</strong>ile shows swelling in the left maxilla.<br />

Figure 5: Orthopantomogram shows poorly defined<br />

radiolucent lesion between canine <strong>and</strong> second premolar in left<br />

maxilla <strong>and</strong> well defined cystic cavities in symphysis at left<br />

body <strong>and</strong> right angle <strong>of</strong> m<strong>and</strong>ible.<br />

Figure 3: Intraoral photograph shows swelling in alveolar<br />

mucosa extending from the central incisor to second<br />

premolar.<br />

DISCUSSION<br />

Hyperparathyroidism is a metabolic bone disease.<br />

Primary Hyperparathyroidism is a disease, in which the<br />

parathyroid gl<strong>and</strong> secretes excessive quantities <strong>of</strong><br />

parathormone (PTH), due to increased activity <strong>of</strong> the<br />

gl<strong>and</strong> due to (1) hyperplasia <strong>of</strong> the gl<strong>and</strong>, (2) adenoma<br />

<strong>of</strong> the gl<strong>and</strong>, (3) functional carcinoma <strong>of</strong> the<br />

parathyroid. Secondary hyperparathyroidism is<br />

secondary to chronic renal failure, rickets <strong>and</strong><br />

osteomalacia [1].<br />

Figure 6: Radiographs <strong>of</strong> long bones shows reduced density <strong>of</strong><br />

bones suggestive <strong>of</strong> osteoporosis.<br />

Hyperparathyroidism is a relatively rare disease which<br />

is three times more common in women than men. It<br />

usually occurs in middle age, but might occur<br />

occasionally in children or in later life. Clinically, pathologic<br />

IJCRI – <strong>International</strong> <strong>Journal</strong> <strong>of</strong> <strong>Case</strong> <strong>Reports</strong> <strong>and</strong> <strong>Images</strong>, Vol. 3 No. 8, August 201 2. ISSN – [0976-31 98]


IJCRI 201 2;3(8):51 –56.<br />

www.ijcasereports<strong>and</strong>images.com<br />

Aswath et al. 54<br />

Figure 7: Photomicrograph shows connective tissue with<br />

plump fibroblasts, multinucleated giant cells <strong>and</strong> blood<br />

vessels (H & E, X400).<br />

Figure 9: Postoperative photograph shows disappearance <strong>of</strong><br />

swelling in the region <strong>of</strong> the left maxilla.<br />

Figure 8: Nuclear scan shows functioning parathyroid lesion<br />

in the region <strong>of</strong> the lower pole <strong>of</strong> the left lobe <strong>of</strong> thyroid.<br />

fractures may be the first symptom <strong>of</strong> the disease,<br />

although bone pain <strong>and</strong> joint stiffness are frequently<br />

heard early symptoms. Urinary tract stone is also a<br />

significant early finding [2].<br />

Intraorally, the first sign <strong>of</strong> the disease may be a<br />

giant cell tumor (Brown tumor) or a cyst <strong>of</strong> the jaw.<br />

Brown tumor represents a giant cell reparative reaction.<br />

The loss <strong>of</strong> phosphorous <strong>and</strong> calcium results in<br />

generalized osteoporosis with attempts to repair the<br />

bone by new bone formation. The new bone may be<br />

resorbed <strong>and</strong> the resorption may lead to pseudocyst<br />

formation. Then the proliferation <strong>of</strong> granulation tissue<br />

from the cystic cavity occurs. As the area <strong>of</strong> bone<br />

resorption undergoes replacement by fibrous tissue,<br />

hemorrhage may occur. Small multinucleated giant cells<br />

may appear in an attempt to remove the blood.<br />

Hemosiderin in blood gives the lesion a brown color.<br />

Malocclusion caused due to sudden drifting <strong>of</strong> teeth<br />

may be the first sign <strong>of</strong> the disease [3­7, 8, 11, 12­14].<br />

Radiographs show the presence <strong>of</strong> small or large<br />

sharply defined radiolucencies suggestive <strong>of</strong> cysts in the<br />

Figure 10: Postoperative intraoral view shows regression <strong>of</strong><br />

intra­oral swelling.<br />

maxilla or m<strong>and</strong>ible. Some lesions show the classical<br />

ground glass appearance. The lamina dura around the<br />

teeth may be partially lost. Multifocal involvement <strong>of</strong><br />

other bones in the body might also be seen [8, 11, 12].<br />

The serum calcium is raised as high as 12 to 20 mg per<br />

100 mL, while the serum phosphorus is lowered to 2 mg<br />

or less per 100 mL. If bone lesions are present serum<br />

alkaline phosphatase <strong>and</strong> serum parathormone levels<br />

IJCRI – <strong>International</strong> <strong>Journal</strong> <strong>of</strong> <strong>Case</strong> <strong>Reports</strong> <strong>and</strong> <strong>Images</strong>, Vol. 3 No. 8, August 201 2. ISSN – [0976-31 98]


IJCRI 201 2;3(8):51 –56.<br />

www.ijcasereports<strong>and</strong>images.com<br />

Aswath et al. 55<br />

regress if the pathology in the parathyroid gl<strong>and</strong> is<br />

corrected.<br />

*********<br />

Figure 11: Postoperative intraoral occlusal <strong>and</strong> periapical<br />

radiographs showing disappearance <strong>of</strong> the radiolucent lesion<br />

between canine <strong>and</strong> second premolar.<br />

Author Contributions<br />

Nalini Aswath – Substantial contributions to conception<br />

<strong>and</strong> design, Acquisition <strong>of</strong> data, Analysis <strong>and</strong><br />

interpretation <strong>of</strong> data, Drafting the article, Revising it<br />

critically for important intellectual content, Final<br />

approval <strong>of</strong> the version to be published<br />

Pravda Chidambaranathan – Analysis <strong>and</strong><br />

interpretation <strong>of</strong> data, Revising it critically for<br />

important intellectual content, Final approval <strong>of</strong> the<br />

version to be published<br />

Guarantor<br />

The corresponding author is the guarantor <strong>of</strong><br />

submission.<br />

Conflict <strong>of</strong> Interest<br />

Authors declare no conflict <strong>of</strong> interest.<br />

Copyright<br />

© Nalini Aswath et al. 2012; This article is distributed<br />

under the terms <strong>of</strong> Creative Commons Attribution 3.0<br />

License which permits unrestricted use, distribution<br />

<strong>and</strong> reproduction in any means provided the original<br />

authors <strong>and</strong> original publisher are properly credited.<br />

(Please see www.ijcasereports<strong>and</strong>images.com<br />

/copyright­policy.php for more information.)<br />

Figure 12: Postoperative orthopantomogram shows<br />

disappearance <strong>of</strong> radiolucent lesion in the left maxilla,<br />

symphysis, left body <strong>and</strong> right angle <strong>of</strong> m<strong>and</strong>ible.<br />

are usually, raised, <strong>and</strong> urinary output <strong>of</strong> calcium is<br />

considerably increased [11, 12]. Histologically,<br />

multinucleated osteoclast like giant cells bone lesions<br />

show lying in hemorrhagic fibrous tissue. Deposits <strong>of</strong><br />

hemosiderin may be seen. Multiple cystic cavities are<br />

noted [5, 6].<br />

CONCLUSION<br />

To conclude hyperparathyroidism is a metabolic<br />

bone disorder that might occur either due to<br />

hypertrophy (or) adenoma <strong>of</strong> the parathyroid gl<strong>and</strong>,<br />

that manifests intraorally as a central gint cell<br />

granuloma. Timely diagnosis, <strong>and</strong> treatment <strong>of</strong> the<br />

parathyroid lesion, results in total regression <strong>of</strong> the<br />

intraoral lesion on its own [10]. Further progression <strong>of</strong><br />

osteoporosis <strong>and</strong> pathological fractures can also be<br />

prevented. Therefore if any case <strong>of</strong> central giant cell<br />

granuloma involving the jaw bones is reported, it is<br />

m<strong>and</strong>atory to rule out hyperparathyroidism. There is no<br />

need to surgically excise the lesion as the lesion will<br />

REFERENCES<br />

1. Neville BW, Damm DD, Allen CM, Bouquot JE. Oral<br />

<strong>and</strong> maxill<strong>of</strong>acial pathology. Philadelphia, W. B.<br />

Saunders 1995.<br />

2. Sapp JP, Eversole LR, Wysocki GP. Contemporary<br />

oral <strong>and</strong> maxill<strong>of</strong>acial pathology. Mosby; St. Louis<br />

1997.<br />

3. Odell EW, Morgan PR. Biopsy pathology <strong>of</strong> the oral<br />

tissues. London; Chapman & hall Medical 1998.<br />

4. Anderson L, Fejerskov O, Philipsen HP. Oral giant<br />

cell granulomas: a clinical <strong>and</strong> histological study <strong>of</strong><br />

129 new cases. Acta Pathol Microbiol Sc<strong>and</strong><br />

1973;81:6.<br />

5. Ficarra G, Kaban LB, Hansen LS. Giant cell lesions<br />

<strong>of</strong> the jaws: a clinicopathologic cytometric study.<br />

Oral Surg Oral Med Oral Pathol 1987;64:44–9.<br />

6. Auclair PJ, Cuenin P, Kratochvil FJ, Slater LJ, Ellis<br />

GL. A clinical <strong>and</strong> histomorphologic comparison <strong>of</strong><br />

the central giant cell granuloma <strong>and</strong> the giant cell<br />

tumor. Oral Surg Oral Med Oral Pathol<br />

1988;66:197–208.<br />

7. Eisenbud L, Stern M, Rothberg M, Sachs SA. Central<br />

giant cell granuloma <strong>of</strong> the jaws: experience in the<br />

management <strong>of</strong> thirty­seven cases. J Oral Maxill<strong>of</strong>ac<br />

Surg 1988;46:37.<br />

8. Whitaker SB, Waldron CA. Central giant cell lesions<br />

<strong>of</strong> the jaws: a clinical, radiologic <strong>and</strong> histopathologic<br />

study. Oral Surg Oral Med Oral Pathol 1993;75:199.<br />

IJCRI – <strong>International</strong> <strong>Journal</strong> <strong>of</strong> <strong>Case</strong> <strong>Reports</strong> <strong>and</strong> <strong>Images</strong>, Vol. 3 No. 8, August 201 2. ISSN – [0976-31 98]


IJCRI 201 2;3(8):51 –56.<br />

www.ijcasereports<strong>and</strong>images.com<br />

Aswath et al. 56<br />

9. Burkes EJ Jr White RP Jr. A peripheral giant cell<br />

granuloma manifestation <strong>of</strong> primary<br />

hyperparathyroidism; report <strong>of</strong> case. J Am Dent<br />

Assoc1989:118:62–5.<br />

10. Peterson CR Burns J.Mowat E. Long term follow­up<br />

<strong>of</strong> untreated primary hyperparathyroidism.Br.Med.j<br />

1984;298:1261–3.<br />

11. Giansanti JS,Waldron CA. Peripheral giant cell<br />

granuloma:review <strong>of</strong> 720 cases.JOral Surgery<br />

1969;27:787–91.<br />

12. Potter BJ, Tiner BD. Central giant cell granuloma.<br />

Report <strong>of</strong> a case. Oral Surg Oral Med Oral Pathology<br />

1993:75:286–9.<br />

13. Shanmugham MS,AI H<strong>and</strong>y SF.Hperparathyroidism<br />

with osteitis fibrosa cystic in maxilla. J. Laryngeal<br />

Otolaryngol 1984;98:417–20.<br />

14. Mundy GR, Cove DH, Fisken R.Primary<br />

hyperparathyroidism;changes in the pattern <strong>of</strong><br />

clinical presentation .Lancet 1980:1:1317–20.<br />

IJCRI – <strong>International</strong> <strong>Journal</strong> <strong>of</strong> <strong>Case</strong> <strong>Reports</strong> <strong>and</strong> <strong>Images</strong>, Vol. 3 No. 8, August 201 2. ISSN – [0976-31 98]

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