Otolaryngology Case Reports 9 (2018) 23–25
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Giant cell reparative granuloma of the nasal cavity in an elderly man: A case report and literature review
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Mohammed Abdel-Rahim Edward Via College of Osteopathic Medicine, Carolinas Campus, United States
ARTICLE INFO
ABSTRACT
Keywords: Giant cell reparative granuloma (GCRG) Giant cell tumor (GCT)
Giant cell reparative granuloma (GCRG) is an uncommon and benign reactive tumor which was first described in the 1950s. It is a reactive tumor that is believed to occur after trauma or inflammation. The lesion is considered central if there is involvement of bone or peripheral if affecting only the soft tissue, such as gingiva or mucosa. It is most commonly found in the bones of the maxilla and mandible, and rarely affects the nasal cavity. It is often seen in children and young adults in the second to third decade of life and it is more common in females. Histopathological classification of GCRG shows a large number of osteoclast-like, multinucleated giant cells within a background of mononuclear stromal cells and spindle-shaped fibroblasts associated with areas of hemorrhage. It is important to distinguish GCRG from giant cell tumors (GCT) since GCTs have a high rate of recurrence, can metastasize, and undergo malignant transformation. Non-surgical treatments of GCRG have been reported in the literature including the use of corticosteroid injections, calcitonin, interferon alpha, radiation therapy, intravenous bisphosphonates, thermal sterilization using laser or cryoprobe, and partial resection. However, surgical resection remains the mainstay of treatment of GCRG to ensure a low chance of recurrence. A case of a left-sided nasal mass that arose from the mucosa of the nasal cavity in an elderly gentleman was removed with surgical resection. Subsequent histopathology yielded the diagnosis of Giant Cell Reparative Granuloma.
Introduction Giant cell reparative granuloma (GCRG) was first described by Jaffe in 1953 [1]. The cause of GCRG is uncertain, but it is classified as a reactive tumor since it is believed to occur after trauma or inflammation. The incidence of this tumor is low with very few cases reported in the literature. When it does occur, it is seen often in children and young adults in the second to third decade of life, with a female predominance. It does not display malignant characteristics, though it can be locally aggressive. The lesion can be grouped into either the central or peripheral categories. It is considered central if there is involvement of bone and peripheral if it only involves the soft tissues (ex: gingiva or mucosa). It mainly occurs centrally, with the predominant location being in the bones of the mandible and maxilla. The second most common site for this tumor is in the bones of the hands and feet [2]. The literature suggests that this tumor is rarely found in the nasal cavity, paranasal sinuses, or the orbit. However, the literature also suggests that when this tumor occurs in the nasal cavity, paranasal sinuses, and the orbit, it can extend into the cranium [3]. Ishinaga et al. report the first case of GCRG of the nasal cavity with intracranial extension [2]. In this paper, this case is of a mass found in the left-sided nasal cavity
which was surgically excised and subsequently found to be a GCRG after pathological confirmation. Case report A 70-year-old man was referred to outpatient otolaryngology with a left sided nasal mass. The left sided nasal mass had grown slowly over the course of 2–3 months. The patient reported nasal congestion and nasal obstruction as symptoms. He denied any episodes of epistaxis, previous history of head and neck cancer, previous history of any nasal mass or tumor. Review of systems was unremarkable. The patient's past medical history included type II diabetes, obstructive sleep apnea, dyslipidemia, glaucoma, and paroxysmal atrial fibrillation. Past surgical history included cholecystectomy, appendectomy, glaucoma surgery, and a renal biopsy. Nasal endoscopy was performed which showed that the right nasal cavity was normal appearing and the scope was advanced to the level of the vocal cords without difficulty. Left sided nasal endoscopy could not be performed due to the size of the mass at the nasal vestibule. There was no external deformity to the nose present. Cranial nerves II-XII were intact. No other pertinent physical exam findings were present. Laboratory data displayed a normal white
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[email protected]. https://doi.org/10.1016/j.xocr.2018.10.002 Received 25 July 2018; Received in revised form 3 September 2018; Accepted 24 October 2018 Available online 25 October 2018 2468-5488/ © 2018 Elsevier Inc. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/BY-NC-ND/4.0/).
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Fig. 2. Histopathology of the Giant Cell Reparative Granuloma of the nasal cavity. Figure A: Low power view of the lesion, with normal squamous epithelium on the right with underlying lesion to the left. Figure B: Ovoid histiocyte-like cells in fibrous stroma with numerous multinucleated giant cells. Figure C: Lesion with associated woven metaplastic bone shown in the lower left figure. Figure D: High power view illustrating the multinucleated giant cells and low mitotic activity of GCRG.
Fig. 1. Preoperative CT scan of a 70-year-old man presenting with a left sided nasal mass. CT scan shows a soft tissue density within the left nasal passageway measuring around 1.5 cm in greatest dimension. Figure A: Axial View. Figure B: Coronal View. Figure C: Sagittal View.
blood cell count of 10.3, hemoglobin of 13.2, hematocrit of 41.4, and a platelet count of 214. All other laboratory data were within normal limits. A preoperative CT scan was performed (Fig. 1) which showed soft tissue density within the left nasal passageway measuring up to 1.5 cm in greatest dimension. There was no associated bony erosion of the nasal bone or septum. There were no associated calcifications. No preoperative biopsy was performed. The decision was made to proceed to the operating room for surgical removal. The mass was found to be protruding off the septum and was excised without difficulty. The nasal airway was patent after excision and no other masses or lesions were identified. The septal mucosa was sutured where the base of the mass was removed. The specimen was sent to pathology for analysis. The post-surgical pathology was deemed inconclusive. At this time, a sample was sent to an outside head and neck pathologist for review and subsequently a diagnosis of Giant Cell Reparative Granuloma was made (Fig. 2).
maxilla and the mandible [11]. Peripheral GCRG's will tend to occur in females younger than 30 years old [11]. There is no difference between the Central and Peripheral GCRG's in terms of their histology. Histopathologically, a GCRG exhibits a large number of osteoclast-like, multinucleated giant cells within a background of mononuclear stromal cells and spindle-shaped fibroblasts associated with areas of hemorrhage [5]. It is important to distinguish GCRG from its differential diagnoses. The main differential diagnosis of GCRG is a Giant Cell Tumor (GCT) since both of these tumors present similarly clinically and histologically. Unlike GCRG, GCTs have a high rate of recurrence, can metastasize and can undergo malignant transformation [5]. GCRG originates from periosteal connective tissue while GCT originates from bone marrow connective tissue [5]. Although histologically GCRG's are very similar to GCTs, GCRG are distinguished from giant cell tumors on the basis that GCRGs exhibit low mitotic activity while GCTs exhibit a high rate of mitotic activity which explains GCT's malignant potential [6]. Also, GCRGs tend to form cysts and exhibit reactive bone formation as seen in the mandible and the maxilla [6]. GCTs of bone have a worse prognosis than GCRG and require adjunctive radiotherapy in addition to surgical excision [7]. GCRG must also be differentiated from Aneurysmal Bone Cysts (ABC) and brown tumors of hyperparathyroidism. In contrast to brown tumors of hyperparathyroidism, lab values of GCRG show normal levels of serum and urinary calcium, phosphates, and alkaline phosphatase; Aneurysmal Bone Cysts appear microscopically as a honeycomb network of thin-walled cavities filled with blood, which is not seen in GCRG [7]. Treatment of GCRG is mainly surgical through local excision with or without curettage and this will cure about 80% of cases which leaves a recurrence rate of 20% [9,10]. In the literature related to GCRG of the mandible, non-surgical treatments include corticosteroid injections to the lesions, calcitonin, interferon alpha, and radiation therapy [5]. Boedeker et al. have reported cases of GCRG of the jaw which have achieved complete remission after trials of calcitonin [10]. Gupta et al.
Discussion In our case, a 70-year-old gentleman presented with a GCRG in the rare location of the nasal cavity. This rare tumor mainly occurs in the bones of the mandible and the maxilla. GCRG's also tend to occur mainly in children and young adults in the second to third decade of life and more commonly in females [4]. The differential diagnosis of any nasal mass begins with the origin of the lesion. It is well known that a Juvenile Nasopharyngeal Angiofibroma (JNA) originates from around the intra nasal location of the sphenopalatine artery or that an Inverting Papilloma originates from the lateral nasal wall. The mass in our case originated from the anterior septum giving a differential diagnosis of nasal polyp, mucosal melanoma, or another rare anomaly. In general, GCRG is classified according to the location of the lesion; central or peripheral. The lesion is considered central if there is involvement of bone, and peripheral if it affects only the soft tissues such as the gingiva or mucosa [4]. Central GCRG's tend to occur in patients in their first and second decade of life and will tend to involve the 24
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reported a case of GCRG involving the maxilla, and they concluded that radiation therapy is a favorable option of treatment of GCRG in cases where surgical resection is not feasible [9,10]. Williams et al., who report a case of GCRG of the petrous temporal bone, report that partial resection followed by radiotherapy is also an option [7]. Kumar et al. reported that there are cases of GCRG which have been sterilized thermally using laser or cryoprobe [4]. Jamil et al., describe a case of a recurrent sphenoid GCRG at 6 months post-operatively and demonstrated successful repeat endoscopic resection without subsequent recurrence at 6 months follow up of the repeat resection [5]. Regarding GCRG of the nasal cavity, Ishinaga, et al., reported having tried oral steroids (30 mg prednisolone) due to the patients rapidly growing tumor, but this steroid treatment was ineffective and the patient eventually required surgical resection [2]. Seo et al., report a case of GCRG of the nasal cavity in a pediatric patient, and they concluded that complete surgical resection is the only definitive form of treatment for GCRG [6]. Seo et al., made this conclusion since other modalities such as corticosteroids, interferon alfa, radiation, and calcitonin traditionally lead to recurrence of the tumor and a need for additional surgery [6]. Felsberg et al., report a case of a frontoethmoidal GCRG and they also are in agreement that surgical resection is the modality of choice for treating this lesion [8]. John et al. report that the treatment of choice for well-defined paranasal sinus lesions is enbloc resection and excision [11]. It can therefore be concluded that based on the literature and from this experience that osseous or nonosseous cases of GCRG should proceed with surgical resection for treatment as this is the only modality that has proven to be definitive.
Funding None. Acknowledgments Mayo Clinic Department of Pathology. Appendix A. Supplementary data Supplementary data to this article can be found online at https:// doi.org/10.1016/j.xocr.2018.10.002. References [1] Jaffe HL. Giant-cell reparative granuloma, traumatic bone cyst, and fibrous (fibroosseous) dysplasia of the jawbones. Oral Surg Oral Med Oral Pathol 1953;6(1):159–75. [2] Ishinaga Hajime, Otsu Kazuya, Mouri Genshin, Takeuchi Kazuhiko. Aggressive giant cell reparative granuloma of the nasal cavity. Case Reports in Otolaryngology, 2013. 2013:690194. 3 pages. [3] Lingaiah J, Ganji L, Yella S, Thatikonda K. A rare case of reparative granuloma of nasal cavity. Int J Otolaryngol Head Neck Surg 2014;3:293–7. [4] Kumar KAJ, Humayun S, Kumar BP, Rao JB. Reparative giant cell granuloma of the maxilla. Ann Maxillofac Surg 2011;1(2):181–6. [5] Jamil OA, Lechpammer M, Prasad S, Litvack Z, Dunn IF. Giant cell reparative granuloma of the sphenoid: case report and review of the literature. Surg Neurol Int 2012;3:140. [6] Seo ST, Kwon KR, Rha K-S, Kim S-H, Kim YM. Pediatric aggressive giant cell granuloma of nasal cavity. Int J Surg Case Rep 2015;16:67–70. [7] Williams JC, Thorell WE, Treves JS, Fidler ME, Moore GF, Leibrock LG. Giant cell reparative granuloma of the petrous temporal bone: a case report and literature review. Skull Base Surg 2000;10(2):89–93. [8] Felsberg GJ, Tien RD, McLendon R. Frontoethmoidal giant cell reparative granuloma. AJNR 1995;16:1551–4. [9] Gupta A, Agrawal SR. Giant cell reparative granuloma of maxilla. Indian J Otolaryngol Head Neck Surg 1999;51(1):29–32. [10] Boedeker CC, Kayser G, Ridder GJ, Maier W, Schipper J. Giant-cell reparative granuloma of the temporal bone: a case report and review of the literature. Ear Nose Throat J 2003;82(12):926–9. 933-4, 936-7. [11] John Mary, Gaurav A, Prabhu J, Sunithi A, Sunithi M, Kurien M. Peripheral giant cell reparative granuloma of the sino-orbital region—a giant lesion: a rare case report. Otorhinolaryngol Clin Int J 2015;6(3):109–13.
Conclusion This is a rare case of a left sided anterior septal mucosal lesion in an elderly man which was surgically excised. This mass was subsequently diagnosed histopathologically as a Giant Cell Reparative Granuloma. Conflicts of interest None.
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