|Year : 2020 | Volume
| Issue : 6 | Page : 3142-3146
Coexistence of hyperparathyroidism and peripheral giant cell granuloma of the jaw: A rare case report
Naina Pattnaik1, Jagadish P Rajguru2, Samarjeet J Pattanaik1, Debajyoti Bardhan3, Bikash Nayak3, Md Mustaq Fizur Islam4
1 Department of Periodontology, Hi-Tech Dental College and Hospital, Bhubaneswar, Odisha, India
2 Department of Oral and Maxillofacial Pathology, Hi-Tech Dental College and Hospital, Bhubaneswar, Odisha, India
3 Department of Oral Medicine and Radiology, Hi-Tech Dental College and Hospital, Bhubaneswar, Odisha, India
4 BDS,PGCE, Genercal Dental Practioner, India
|Date of Submission||27-Mar-2020|
|Date of Decision||25-Apr-2020|
|Date of Acceptance||11-May-2020|
|Date of Web Publication||30-Jun-2020|
Dr. Naina Pattnaik
Department of Periodontology, Hi-Tech Dental College and Hospital, Bhubaneswar, Odisha
Source of Support: None, Conflict of Interest: None
Peripheral giant cell granuloma (PGCG) known as “giant cell epulis” is a benign, reactive exophytic gingival lesion that accounts for less than 10% of all gingival lesions. PGCG affects females more than males with middle age predilection. Till now the etiology of PGCG remains unclear but various factors that can cause PGCG include poor oral hygiene, food impaction, following an extraction, dry mouth, hormonal disturbance, and hyperparathyroidism. The reported recurrence rate of the lesion is 5.0%–70.6%. The present case report describes the rare case of PGCG with primary hyperparathyroidism in a male patient with a history of swelling in the mandibular anterior region.
Keywords: Giant cell, hyperparathyroidism, peripheral giant cell granuloma
|How to cite this article:|
Pattnaik N, Rajguru JP, Pattanaik SJ, Bardhan D, Nayak B, Islam MF. Coexistence of hyperparathyroidism and peripheral giant cell granuloma of the jaw: A rare case report. J Family Med Prim Care 2020;9:3142-6
|How to cite this URL:|
Pattnaik N, Rajguru JP, Pattanaik SJ, Bardhan D, Nayak B, Islam MF. Coexistence of hyperparathyroidism and peripheral giant cell granuloma of the jaw: A rare case report. J Family Med Prim Care [serial online] 2020 [cited 2020 Jul 15];9:3142-6. Available from: http://www.jfmpc.com/text.asp?2020/9/6/3142/287895
| Introduction|| |
Peripheral giant cell granuloma (PGCG) is a common benign, reactive exophytic lesion of the oral mucosa. It can arise from periodontal ligament or periosteum of the alveolar bone. There are various etiological factors for PGCG such as poor oral hygiene, food impaction, following the extraction, xerostomia, hormonal imbalance, and hyperparathyroidism (HPT)., The recurrence rate of the lesion is 5.0%–70.6%. In the case of recurrence of any lesion, multiple etiologies should be considered. PGCG is considered to be a reactive lesion occurring due to chronic irritation and shares an identical morphology with central giant cell granuloma. The genetic etiology includes activating mutations in the MAP-kinase signaling pathway, KRAS, and FGFR1 mutations.
In giant cell lesions, patients should be evaluated for HPT to rule out a brown tumor. HPT was first found by Von Recklinghausen in 1891. It is of 3 types: primary, secondary, and tertiary. The prevalence of primary hyperparathyroidism (PHPT) associated with giant cell lesions is 5.9%. PHPT results in hypercalcemia and affects many organs like bone, kidney, soft tissues, and central nervous system. The odontogenic tissues and jawbones are also affected as a systemic manifestation of PHPT. The occurrence of both PGCG and PHPT is more in females. The ratio of female and male for PGCC is 2:1 and for PHPT is 3:1., Hereby, we are presenting a rare case of PGCG with PHPT in a male patient.
| Case Report|| |
A 40-year-old male patient with a chief complaint of swollen gums in the anterior region of the mandible reported to the Department of Periodontics and Implantology of Hi-Tech Dental College and Hospital, Bhubaneswar. The patient had the same history of swollen gums 2 years back which was excised. Following that after a year, there was a small swelling developed in relation to #32, #33. The swelling grew rapidly and attained a size of 5 × 4 cm [Figure 1] extending from #32 to #33. The swelling was well-defined solitary, oval in size with a pedunculated base. The surface was smooth and shiny with surface ulcerations. The etiology of the present lesion was found to be trauma; thus, the diagnosis was more toward pyogenic granuloma. Further on palpation, the swelling was found to be soft and nontender and there was profuse bleeding on probing. The oral hygiene status of the patient was fair with mild calculus present around the anterior mandibular teeth. Although there was no other abnormality detected on the extraoral examination, the patient looked weak and emaciated. Also, he complained of occasional fatigue and tiredness. A provisional diagnosis of pyogenic granuloma was made. Following that, a routine lab investigation was carried out, which revealed high serum calcium (10.9 mg/dl) and PTH level (72 pg/ml). A diagnosis of PHPT was made based on the findings. The patient was referred to the endocrinologist for further treatment. Further intraoral investigation radiographic investigation showed mild crestal bone loss in relation to # 32, #33. Written informed consent was obtained from the patient along with the ethical clearance from the institutional ethical committee (E/C3456). Complete surgical excision of the swelling was performed after scaling and root planning [Figure 2]. The periodontal dressing was placed over the surgical site [Figure 3]. The patient was prescribed with antibiotics and analgesics and oral hygiene instructions were given. The histopathology report confirmed the diagnosis of peripheral giant cell granuloma. The H and E section showed para keratinized stratified squamous epithelium covering underlying granulomatous stroma composed of numerous proliferating multinucleated giant cells, fibroblasts, proliferating vessels, and chronic inflammatory cells [Figure 4]a and [Figure 4]b. A 6-month and 1-year follow-up showed no further swelling in the oral cavity [Figure 4] and [Figure 5].
|Figure 3: The periodontal dressing (Coe-Pak) was placed over the surgical site|
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|Figure 4: One-year postoperative follow-up. a: Histopathological picture showing para keratinized epithelium overlying the connective tissue stroma. b: Giant cells in the stroma|
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| Discussion|| |
This case report represents a case of PGCC with PHPT. PGCG is a benign gingival lesion which reckoned less than 10% of all gingival lesions. Jaffe coined the term giant cell reparative granuloma for PGCG. Further, Bernier and Cahn named it peripheral and central cell reparative granuloma. Subsequently, the giant cell granuloma was divided as central and peripheral by Bhaskar et al. in the year 1959. Peripheral giant cell granuloma is a benign, nonodontogenic tumor of the oral cavity. It mostly occurs in the mandible and has female predilection. PGCG is more common in premolar and molar region though its occurrence in incisor and the canine region has been reported., Usually, PGCG is well-demarcated, sessile, or pedunculated deep red to bluish red in color similar to the present case.
Subramanian et al. reported a case of peripheral giant cell granuloma in a 38-year-old male that represented as a localized painless, lobulated, purplish-red overgrowth of gingiva localized to the region of 14 and 15, extending from the interdental papilla on the buccal aspect to the palatal aspect.
The differential diagnosis of PGCG includes pyogenic granuloma, hemangioma, metastatic carcinoma, and peripheral ossifying fibroma, which are proliferative gingival lesions that can show very similar characteristics but have a difference in histology and recurrence rate., Another condition could also be considered such as peripheral odontogenic fibroma that represents clinically a dome-shaped or nodular, fibrotic growth in gingiva like PGCG. However, histologically it consists of fibrous or fibromyxomatous stroma containing varying numbers of islands of odontogenic epithelium, which are distinguishable from PGCG. The striking histopathological features of PGCG are the presence of numerous proliferating fibroblasts, vascularized fibro cellular stroma with numerous capillaries, and numerous multinucleated giant cells., The distinctive feature of PGCG is mainly due to the excess number of giant cells that are clustered in the connective tissue stroma [Figure 6]. The exact origin of giant cells is uncertain but it has been suggested that cells like osteoblasts, macrophages, endothelial cells, and spindle cells can give rise to these multinucleated giant cells.
If there were multiple lesions or if recurrence of the same lesions occurred after surgical removal, then other conditions should also be considered in differential diagnosis such as brown tumor of HPT, cherubism, and aneurysmal bone cyst. Brown tumor can perforate the cervical region of the tooth, and aneurysmal bone cyst is affecting the bone with a more aggressive nature. PHPT is considered as an endocrine disorder that results due to the autonomous overproduction of PTH, usually resulting from parathyroid adenoma (90%), parathyroid hyperplasia (3%), or less commonly an adenocarcinoma (3%), and rarely associated with Noonan type syndrome.
In the present case, as there was a history of multiple excision and recurrence of the lesion, speculation for further lab investigations was carried out to rule out other diseases. Based on clinical findings and histological and lab investigations, a diagnosis of PGCG with PHPT was established. The patient presenting with PGCG may also have other signs and symptoms of PHPT. Generalized weakness, anemia, gastric ulcer, renal stones, and osteoporosis are features of PHPT. In our case, the patient also complained of continuous weakness and his appearance was also very weak and emaciated; thus, systemic involvement needed to be evaluated. Smith et al. reported a similar case of PHPT in which the initial clinical presentation was that of an intraoral lesion and PHPT was discovered on routine blood analysis. Also, Burkes reported a case of peripheral giant cell granuloma with the manifestation of PHPT. Parbatani et al. presented a case of giant cell epulis as an initial feature of PHPT. Vendrell Marques et al. and Matinz-Gavidia et al. reported cases of the maxillary brown tumor as an early sign of PHPT. Also, two cases of giant cell lesions were established at the discovery of PHPT by Aoune et al. Choi et al. found a case of PGCG associated with HPT secondary to end-stage renal diseases. Both PGCG and PHPT have female predilection with female to male ratio; for PGCG, it is 2:1 and for PHPT, it is 3:1.,, However, various cases had been reported affecting the male gender more. Chaparro-Avendano et al. reported a study involving three males with PGCG. Bhaskar et al., Salum et al., Zhang et al., and Murat et al. showed male predominance in PGCG. Also, Mazeh et al. compared the role of gender in PHPT and they concluded that male patients present without symptoms. The positive influence of estrogen and progesterone inclined toward hormonal etiology in our case along with the presence of secondary local factors such as plaque and calculus. There were no classical symptoms of hypercalcemia in our patients, such as bone fractures, renal stones, and abdominal groans. Other symptoms also include paresthesia, headaches, recent fractures, constipation, polyuria, and polydipsia. Mostly the symptoms were asymptomatic and diagnosed while routine laboratory investigations. The treatment of the PGCG is complete excision of the lesion along with the curettage of the base and borders of the lesion. In the present case, complete excision of the lesion was done along with thorough curettage.
Implications for clinical practice
The pathologist may come across many inflammatory conditions in response to dental plaque like gingivitis and periodontitis. Lesion such as PGCG has greater chances of recurrences and this must be kept in mind while treating such pathologies. The treatment of PGCG comprises complete surgical resection along with the entire base of the lesion. The underlying source of irritation should also be eradicated while treating such lesions to prevent a recurrence., Thus, to reach an accurate diagnosis, clinical history, histological, radiological, and laboratory investigations are necessary. The dentist should be aware of the possible association of oral lesions with systemic diseases. The biochemical tests that are diagnostic for such systemic diseases should be recommended to rule out the associated conditions.
Summary and Conclusion
Histopathological examination along with biochemical tests is essential to predict the conclusive diagnosis and treatment planning. The present case confirmed the diagnosis of PGCG with PHPT. Thus, it is necessary that such oral lesions with ambiguous causes should be attributed to a specialist for accurate diagnosis and further treatment.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that his name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Tandon PN, Gupta SK, Jurel SK, Saraswat A. Peripheral giant cell granuloma. Contemp Clin Dent 2012;3:S118-21.
Motamedi MH, Eshghyar N, Jafari SM, Lassemi E, Navi F, Abbas FM, et al
. Peripheral and central giant cell granulomas of the jaws: A demographic study. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2007;103:e39-43.
Eversole LR, Rovin S. Reactive lesions of the gingiva. J Oral Pathol 1972;1:30-8.
Reichart PA, Philipsen HP. Gingiva. In: Reichart PA, Philipsen HP, editors. Color Atlas of Dental Medicine. Oral Pathology. New York: Thieme; 2000. p. 148-75.
Cleven AHG, Schreuder WH, Groen E, Kroon HM, Baumhoer D. Molecular findings in maxillofacial bone tumours and its diagnostic value. Virchows Archiv 2020;476:159-74.
Magalhães DP, Osterne RL, Alves AP, Santos PS, Lima RB, Sousa FB. Multiple brown tumours of tertiary hyperparathyroidism in a renal transplant recipient: A case report. Med Oral Pathol OralCirBucal; 2010;15:e10-3.
Triantafillidou K, Zouloumis L, Karakinaris G, Kalimeras E, Iordanidis F. Brown tumors of the jaws associated with primary or secondary hyperparathyroidism. A clinical study and review of the literature. Am J Otolaryngol 2006;27:281-6.
Rai S, Rattan V, Bhadada SK. Giant cell lesions associated with primary hyperparathyroidism. J Maxillofac Oral Surg 2015;14:930-4.
Jaffe HL. Giant-cell reparative granuloma, traumatic bone cyst, and fibrous (fibro-osseous) dysplasia of the jawbones. Oral Surg Oral Med Oral Pathol 1953;6:159-75.
Silverman S Jr, Ware WH, Gillooly C Jr. Dental aspects of hyperparathyroidism. Oral Surg Oral Med Oral Pathol 1968;26:184-9.
Silverman S Jr, Gordan G, Grant T, Steinbach H, Eisenberg E, Manson R. The dental structures in primary hyperparathyroidism. Studies in forty-two consecutive patients. Oral Surg Oral Med Oral Pathol 1962;15:426-36.
Bernier JL, Cahn LR. The peripheral giant cell reparative granuloma. J Am Dent Assoc 1954;49:141-8.
Bhaskar SN, Bernier JL, Godby F. Aneurysmal bone cyst and other giant cell lesions of the jaws: Report of 104 cases. J Oral SurgAnesthHosp Dent Serv 1959;17:30-41.
Yadalam U, Bhavya B, Kranti K. Peripheral giant cell granuloma: A case report. Int J Dent Case Rep 2012;2:30-4.
Shafer WG, Hine MK, Levy BM. A Textbook of Oral Pathology. 4th
ed. Philadelphia: WB Saunders Company; 1983. p. 185-6.
Kaya GS, Yalcýn E, Tozoðlu U, Þipal S, Demirci E. Huge peripheral giant cell granuloma leading to bone resorption: A report of two cases. Cumhuriyet Dent J 2011;14:219-24.
Subramanian P, Kharbuli D, Das SJ. Peripheral giant cell granuloma: A case report. J Res Med Dent Sci 2019;7:169-72.
Mishra MB, Bhishen KA, Mishra S. Peripheral ossifying fibroma. J Oral Maxillofac Pathol 2011;15:65-8.
] [Full text]
Patil VA, Shivakumar TP. Oral Pyogenic granuloma: A report of two cases. Ann Essences Dent 2010;2:93-7.
Daley TD, Wysocki GP. Peripheral odontogenic fibroma. Oral Surg Oral Med Oral Pathol 1994;78:329-36.
Mighell AJ, Robinson PA, Hume WJ. Peripheral giant cell granuloma: A clinical study of 77 cases from 62 patients, and literature review. Oral Dis 1995;1:12-9.
Adlakha VK, Chandna P, Rehani U, Rana V, Malik P. Peripheral giant cell granuloma. J Indian Soc Pedod Prev Dent 2010;28:293-6.
] [Full text]
Rodrigues SV, Mitra DK, Pawar SD, Vijayakar HN. Peripheral giant cell granuloma: This enormity is a rarity. J Indian Soc Periodontol 2015;19:466-9.
] [Full text]
Moghe S, Gupta MK, Pillai A, Maheswari A. Peripheral giant cell granuloma: A case report and review of literature. People's J Sci Res 2013;6:55-9.
Todero MA, Monaco A, DAmario M, La Carbonara M, Capogreco M. Peripheral giant cell granuloma (giant cell epulis) associated with metabolic diseases: Case report and literature review. Ann Stomatol (Roma) 2013;4:45.
Gosavi S, Kaur H, Gandhi P. Multifocal osteolytic lesions of the jaw as a road map to diagnosis of brown tumor of hyperparathyroidism: A rare case report with review of literature. J Oral Maxillofac Pathol 2020;24(Suppl S1):59-66.
Smith BR, Fowler CB, Svane TJ. Primary Hyperparathyroidism Presenting as a “Peripheral” Giant Cell Granuloma. J Oral Maxillofac Surg 1988;46:65-9.
Burkes ER. A pheripheralgaint –cell granuloma manifestation of primary hyperparathyroidism: Report of a case. J Am Dent Assoc 1989;118;62-4.
Parbatani R, Tinsley GF, Danford MH. Primary hyperparathyroidism presenting as a giant –cell epulis. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1998;85:282-4.
Vendrell Marques JB, Artazkoz del Toro JJ, Faubel Serra M, Campos Dana JJ, Pinon Selles F. Brown tumor of the maxilla as initial manifestation of primary hyperparathyroidism. An Otorrinolaringol Ibero Am 1991;18:301-8.
Martinez-Gavidia EM, Bagan JV, Milian-Masanet MA, Lloria de Miguel E, Perez-Valles A. Highly aggressive brown tumour of the maxilla as first manifestation of primary hyperparathyroidism. Int J Oral Maxillofac Surg 2000;29:447-9.
Aoune S, Khochtail H, Dahdouh C, Turki A, Mokni M, Bakir A. Giant cell lesions of maxilla disclosing primary hyperparathyroidism. Rev Stomatol Chir Maxilllofac 2000;101:86-9.
Choi C, Terzian E, Schneider R, Trochesset DA. Peripheral giant cell granuloma associated with hyperparathyroidism secondary to end-stage renal disease: A case report. J Oral Maxillofac Surg 2008;66:1063-6.
Chaparro-Avendano AV, Berini – Aytes L, Gay-Escoda C. Peripheral giant cell granuloma. A report of five cases and review of literature. Med Oral Patol Oral Cir Bucal 2005;10:53-7; 48-52.
Bhaskar SN, Cutright DE, Beasley JD, III, Perez B. Gaint cell reparative granuloma (peripheral): Report of 50 cases. J Oral Surg 1971;29:110-5.
Salum FG, Yurgel LS, Cherubini K, De Figueiredo MA, Medeiros IC, Nicola FS. Pyogenic granuloma, peripheral giant cell granuloma, and Peripheral ossifying fibroma: Retrospective analysis of 138 cases. Minerva Stomatol 2008;57:227-32.
Zhang W, Chen Y, An Z, Geng N, Bao D. Reactive gingival lesions: A retrospective study of 2,439 cases. Quintessence Int 2007;38:103-10.
Muratakgul H, Gugrmu M, Haroli A. Peripheral giant cell granuloma. A clinical and radiological study. The Pain Clinic 2004;16:59-63.
Mazeh H, Kouniavsky G, Schneider DF, Makris K I, Sippel R S, Dackiw AP, Chen H, Zeiger M A. Intra thyroidal parathyroid glands: Small, but mighty (a Napoleon phenomenon). Surgery 2012;152:1193-200.
Carr ER, Contractor K, Remedios D, Burke M. Can parathyroidectomy for primary hyperparathyroidism be carried out as a day- case procedure? J Laryngol Otol 2006;120:939-41.
Bodner L, Peist M, Gatot A, Fliss DM. Growth potential of peripheral giant cell granuloma. Oral Surg Oral Med Oral Pathol Oral Radiol 1997;83:548-51.
Flaitz CM. Peripheral giant cell granuloma: A potentially aggressive lesion in children. Pediatr Dent 2000;22:232-3.
Abu Gharbyah AZ, Assaf M. Management of a peripheral giant cell granuloma in the esthetic area of upper jaw: A case report. Int J Surg Case Rep 2014;5:779-82.
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]