International Journal of Pediatric Otorhinolaryngology Extra
Volume 1, Issue 4 , Pages 297-300, December 2006

Surgery and calcitonin therapy in childhood central giant cell granuloma

  • Nalan Yazici

      Affiliations

    • Department of Pediatric Oncology, Hacettepe University, Institute of Oncology, 06100 Ankara, Turkey
    • Corresponding Author InformationCorresponding author. Tel.: +90 312 3052990; fax: +90 312 3107018.
    • ,
  • Bilgehan Yalçin

      Affiliations

    • Department of Pediatric Oncology, Hacettepe University, Institute of Oncology, 06100 Ankara, Turkey
    • ,
  • Taner Yilmaz

      Affiliations

    • Department of Otolaryngology–Head & Neck Surgery, Hacettepe University, Faculty of Medicine, 06100 Ankara, Turkey
    • ,
  • Canan Akyüz

      Affiliations

    • Department of Pediatric Oncology, Hacettepe University, Institute of Oncology, 06100 Ankara, Turkey
    • ,
  • Kader Karli Oguz

      Affiliations

    • Department of Radiology, Hacettepe University, Faculty of Medicine, 06100 Ankara, Turkey
    • ,
  • Arzu Sungur

      Affiliations

    • Department of Pathology, Hacettepe University, Faculty of Medicine, 06100 Ankara, Turkey
    • ,
  • Münevver Büyükpamukçu

      Affiliations

    • Department of Pediatric Oncology, Hacettepe University, Institute of Oncology, 06100 Ankara, Turkey

Received 29 July 2006; received in revised form 5 September 2006; accepted 8 September 2006.

Article Outline

Summary 

Central giant cell granuloma is a rare and a benign lesion of the jaws. Conventional treatment is surgery but other approaches including calcitonin are being used in unresectable cases. Herein, we report a case of maxillary tumor causing prominent asymmetry and swelling in the face of a young girl who was effectively treated with surgery following unsuccessful calcitonin therapy.

Keywords: Central giant cell granuloma, Calcitonin, Surgery, Children

 

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1. Introduction 

Central giant cell granuloma (CGCG) is a relatively rare and benign lesion which constitutes 7–17% of all benign tumors affecting the jaws [1], [2]. The incidence of the disease is reported as 0.00011% in a population-based retrospective study [3]. As defined by WHO, it is an intraosseous lesion which comprises cellular fibrous tissue including multiple hemorrhagic aggregations of multinucleated giant cells and presence of trabeculae of wowen bone [4]. Herein, a child with maxillary CGCG who was treated effectively by surgery after unsuccessful calcitonin treatment is reported.

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2. Case 

A 4-year-old, previously healthy girl was admitted with a painless, slowly growing mass on the left side of her face. In her past medical history, interferon alpha 2a was administered at another hospital for about 3 months with a biopsy-proven diagnosis of ‘giant cell mesenchymal tumor’. At initial physical examination pallor and a huge, hemorrhagic mass were detected. The mass was protruding from the mouth and the swelling on her left cheek was causing a compression to the left orbit leading to the closure of the left eye (Fig. 1A). The initial hemoglobin level was 4.8g/dl considered to be the result of frequent bleeding from the mass either spontaneously or during oral intake. Additional laboratory investigations including WBC, platelets and differential count, serum biochemistry and serum parathyroid hormone were normal. Initial magnetic resonance imaging (MRI) which was performed at another institute revealed a left maxillary solid mass occupying ipsilateral maxillary sinus, invading the hard palate, alveolar processes, nasal cavity and the orbit as well as the pterygopalatine fossa (Fig. 2A). On MRI performed after a year of follow-up, the mass was found to have increased in size profoundly with a prominent exophytic portion growing anteroinferiorly (Fig. 2B). Destruction of hard palate, alveolar processes, ethmoidal cells, lamina papyracea, zygoma, orbital walls and pterygoid plates by infiltrating heterogeneous and necrotic mass was displayed on a computed tomography (CT) examination (Fig. 2C). Lymphadenopathies <1cm in size were located at the jugular chain, submandibular and posterior cervical region. Abdominal ultrasonography, thoracic and abdominal CT studies were within normal findings. Histopathological examination of the biopsy revealed ‘central giant cell granuloma’. Total resection of the mass was performed via Weber–Ferguson–Moore incision and sponges containing corticosteroid was applied to the resection cavity. Histopathological diagnosis of the specimen was the same with the initial biopsy. Calcitonin receptor examination was not available. Calcitonin was applied intranasally with a dose of 200IU for 10 months. A surgical reduction in size of the mass especially of the outpouching portion was observed on postoperative CT examination and residual tumor remained stable until termination of calcitonin treatment (Fig. 2D). A second surgery was performed for reconstruction. However, recurrent mass was discovered in the cavity; reconstruction was cancelled and the mass was removed again. Gross total resection was done. The patient is still under follow-up with no evidence of recurrence on neither physical examination nor MRI for 22 months after the cessation of the treatment (Fig. 2E).

  • View full-size image.
  • Fig. 2. 

    (A) Initial MR imaging shows left maxillary enhancing tumor which infiltrates nasal cavity and pterygopalatine fossa on fat-suppressed T1-weighted (T1W) spinecho (SE) (TR/TE; 560/12ms). (B) A postcontast T1W SE imaging obtained at 1 year of diagnosis displays that lesion increased in size profoundly (T1W SE; 675/15ms). (C) The tumor caused massive destruction–infiltration of the maxillary sinus, zygoma, pterygoid plates and lateral nazal wall as shown on contrast-enhanced CT image. (D) Left maxillary residual tumor with a reduction in outpouching portion is seen on CT examination upon termination of the calcitonin treatment. (E) No residual or recurrent mass is observed on postcontrast transverse images (TR/TE; 500/70ms) performed 22 months after termination of hormonal and surgical treatment.

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3. Discussion 

Central giant cell granuloma is first described by Jaffe as a reactive lesion to an intraosseous hemorrhage following trauma to the jaw and it was clinically and histologically distinct from a giant cell tumor [5]. Central tumors usually arise deep in cortex whereas peripheral ones originate in soft tissue outside the cortex. Although in some cases CGCG is proposed to be in association with genetic abnormalities, the etiology is still unclear [6].

Although it is often a disease of adolescents and young adults, children younger than 10 years were also reported previously [1], [7], [8], [9], [10], [11], [12], [13]. Mandible is more frequently involved and in some series female gender is more commonly affected than males [3], [8], [10], [11], [13]. Conventional treatment is curettage and en bloc resection. Besides, in aggressive or unresectable cases; radiotherapy, cryosurgery; local steroid injections, interferon alpha are being used [3], [9], [14], [15], [16], [17], [18]. In our case interferon alpha treatment was applied for a month but the parents refused further treatment after a clinical progression was observed. Some authors reported the successful use of calcitonin in the treatment of CGCG because of the histological similarity of brown tumors which occur in hyperparathyroidism to CGCG lesions [6], [9], [14], [19], [20], [21]. The effect of calcitonin is defined as a direct inhibition on osteoclasts leading to a decrease in bone resorption.

In our case, we used intranasal calcitonin after a total resection. Previously, in 8 of 10 patients who were treated with subcutaneous or intranasal (only one patient) calcitonin for 19–26 months, no recurrence was observed through 26–50 months [9]. In another small series, four pediatric cases treated with subcutaneous or intranasal calcitonin showed no recurrence with a follow-up of 7 months to 3 years [6]. A study of three patients also revealed no recurrence with a median follow-up of 8 months after the cessation of therapy [21]. In our patient, a progressive mass was observed radiologically under calcitonin treatment and hormone therapy was ceased at 9 months. The recurrent lesion was finally resected. The treatment duration of calcitonin might have been shorter in our patient because in a previous report, no radiological change in the lesions under calcitonin treatment for the first 4–6 months was reported [9].

The conventional treatment of CGCG is surgery. Although successful results and few recurrences were reported in several studies [1], [12], [22], [23], [24]; in one of the retrospective studies, disease-free survival at 5 years following surgery was 76.1% [3]. In our patient, a disease-free period of 22 months was obtained after the second gross total resection. Surgery seems to be the gold standard in treatment of central giant cell granuloma. By this case it might be assumed that the combination of calcitonin treatment with surgery appeared to have no beneficial effect on recurrence after the surgery.

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References 

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PII: S1871-4048(06)00087-6

doi:10.1016/j.pedex.2006.09.002

International Journal of Pediatric Otorhinolaryngology Extra
Volume 1, Issue 4 , Pages 297-300, December 2006

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