Multiple recurrent giant cell lesions associated with high circulating levels of parathyroid hormone-related peptide in a young adult

Parathyroid hormone-related peptide (PTHrP, PLP) has previously been identified and assays are now available which can be used in clinical situations. A case is reported of a normocalcaemic young adult female in whom multifocal recurrent giant cell osteolytic lesions in the maxilla and elsewhere were associated with a raised plasma level of parathyroid hormone-related peptide. The lesions were histologically identical to reparative giant cell granuloma of the jaws and to osteitis fibrosa cystica associated with hyperparathyroidism.     

Giant Cell Lesions Associated with Primary Hyperparathyroidism

Aims and Objective

To evaluate the prevalence, clinical features, diagnostic laboratory values and treatment outcome of giant cell lesions (brown tumors) associated with primary hyperparathyroidism (PHPT) in oral and maxillofacial region.

Study Design

A 5 year retrospective data was analyzed wherein all histopathologically proven cases of giant cell lesions involving oral and maxillofacial region were evaluated. Out of these cases, those associated with PHPT were tabulated. Correlation was established with other concomitant clinical features and also with the laboratory values of altered serum calcium, phosphate, alkaline phosphate and parathormone. Follow up of these cases after the correction of PHPT was also noted.

Result

Out of 85 cases of histopathologically proven giant cell lesions, five cases were associated with PHPT. There was involvement of maxilla and mandible in one case each. Only frontal bone was involved in two cases. Fifth case had multiple lytic lesions in maxilla and frontal bone. All patients consistently showed very high values of alkaline phosphate and parathormone. Hypercalcemia and hypophosphatemia was noted in four cases. All cases showed regression of the lytic lesion after parathyroidectomy obviating the need for surgical excision of the jaw lesions.

Conclusion

Giant cell lesions (brown tumors) associated with PHPT in oral and maxillofacial region are rare clinical entities. The prevalence of PHPT associated giant cell lesions is 5.9 %. They are clinically, radiologically and histopathologically similar to any other peripheral or central giant cell tumor. Relevant history may alert the clinician and altered biochemical values may help in correlating the oral and maxillofacial findings with the underlying systemic disease. At times, the brown tumor maybe the only presenting sign leading to the diagnosis of PHPT.     

Radiographic patterns of multiple myeloma in the jawbones of patients treated with intravenous bisphosphonates

Background

The purpose of this study was to evaluate whether intravenous (IV) bisphosphonate (BP) therapy can change the radiographic patterns of multiple myeloma (MM) in the jawbones.

Bilateral maxillary brown tumours as the first presentation of primary hyperparathyroidism

Introduction

Symptomatic skeletal disease in primary hyperparathyroidism is over 30 times more common in India compared to the west. The classical “brown tumour” is commonly seen with the major sites being ends of long bones, the pelvis and ribs. Facial involvement is rare and, when present, usually involves the mandible.

Case report

We report a 68-year-old gentleman with a rare initial presentation of primary hyperparathyroidism with bilateral maxillary brown tumours.

Discussion and conclusion

Successful parathyroid surgery resulted in a regression in the tumours. The report highlights the need to consider primary hyperparathyroidism in the initial differential diagnosis of bony lesions of the jaw.     

Brown tumor of the jaws as a manifestation of tertiary hyperparathyroidism: A literature review and case report

Brown tumor of the jaws is a manifestation of hyperparathyroidism consisting of osteolytic lesions that show proliferation of multinucleated giant cells in the maxilla and/or mandible. Differential diagnosis of these lesions from local central giant‐cell granuloma is mandatory for the correct treatment of the patient. Radiographic and histopathological exams of the jaw lesion are not sufficient to determine the diagnosis, which requires laboratory tests including serum levels of calcium, alkaline phosphatase, parathyroid hormone (PTH) and phosphate, and radiographic examination of other bones as well, such as hand‐wrist, pelvis, and femur. We present here a brief literature review focusing on the clinical and radiographic features, diagnostic criteria and treatment of brown tumor and also report a case of the disease affecting the jaw.     

Differential diagnosis in a case of brown tumor caused by primary hyperparathyroidism

Fibrous-cystic osteitis is a bone metabolic disorder related to hyperparathyroidism. This pathological condition shows a bone catabolism enhancement, due to increased level of PTH. Brown tumour is a uni- or multi-focal bone lesion, which represents the terminal stage of the hyperparathyroidism-dependent bone pathology. This focal lesion is not a real neoplasm showing itself as a cellular reparative process, mainly interesting the jaws, specially the mandible. Because of the similar radiological features (cyst-like radiolucency) showed by other lesions, the diagnosis can be difficult. Histology cannot guarantee a certain diagnosis, some lesions, such as giant cell tumour, giant cell granuloma, aneurysmal bone cyst and cherubism, show a similar macroscopical and microscopical features. Differential diagnosis is possible only by comparative evaluation of clinical, radiological and biochemical evidences. Personal experience with a patient affected by maxillary expansive lesion previously diagnosed as GCT is reported. Radiological examinations showed another cyst-like lesion involving the mandible. Clinical history and multifocality of lesions were suggestive for the presence of a systemic disease, laboratory data allowed a primary hyperparathyroidism diagnosis. Parathyroid scintigraphy was performed and detected a parathyroid adenoma. In first instance the patient underwent to surgical operation on the jaws in order to stop the rapid progression of bone lesions, and then another operation for the removal of parathyroid adenoma was performed.     

Comparison of the ultrastructure of a malignant tumor of the mandible containing giant cells with Paget's disease of bone

Abstract Distinction between lesions of the jaws containing giant cells can be difficult. This study reports the results of the study of the ultrastructure of 30 patients with Paget's disease in comparison with a patient diagnosed as having malignant giant cell tumor of the mandible. The ultrastructure of the malignant giant cell tumor of the mandible was found to contain nuclear inclusions morphologically identical with the 12–15 nm tubules characteristic of the nuclei of osteoclasts in Paget's disease of bone. No clinical or laboratory signs of Paget's disease were found in this patient. A search of the literature failed to produce reports of identical nuclear inclusions in soft tissue giant cell tumors, osteosarcomas, reparative granulomas containing giant cells or in osteitis fibrosa eystica. Such nuclear inclusions have been reported in benign giant cell tumors of bone, one other in malignant giant cell tumor and cases of polymyositis. These observations raise the question of a possible virus associated with the tumor since there arc recent reports of immunological evidence for the presence of a virus in Paget's disease, and viruses have been suspected in polymyositis.     

Radiographic findings of bisphosphonate-related osteomyelitis of the jaw: investigation of the diagnostic points by comparison with radiation osteomyelitis, suppurative osteomyelitis, and diffuse sclerosing osteomyelitis

Objectives

To investigate the possibility of radiographic examination as a tool for differential diagnosis of bisphosphonate-related osteomyelitis of the jaw (BROMJ).

Methods

The radiographic findings of the mandibular lesions in 25 BROMJ patients were compared with those in 36 radiation osteomyelitis, 92 suppurative osteomyelitis, and 34 diffuse sclerosing osteomyelitis patients. The diagnostic criterion for BROMJ was osteomyelitis in patients with experience of bisphosphonate therapy. The amount and duration of bisphosphonate administration and presence and term of bone exposure were not considered for diagnosis.

Results

BROMJ revealed characteristic radiographic findings. Osteolytic change that started at the superficial layer of bone (cortical bone), followed by extension between the cortical and cancellous bone, large cancellous bone sequestrum formation often accompanied by extensive osteolytic change of the cortical bone or fairly large cortical bone sequestrum formation, and solid-type periosteal reaction formed at a site related to the margin of the remaining cortical bone were found to be pathognomonic, and were observed not only in cases with bone exposure but also in cases without bone exposure. A relationship between lamina dura thickening and the lesion was doubtful. Osteosclerotic change was frequent and remarkable, but not specific.

Conclusions

Radiographic examination was considered to be useful for differential diagnosis of BROMJ. Inclusion of the pathognomonic radiographic findings into the definition of BROMJ will make it possible to achieve earlier and more accurate diagnoses. In addition, the active bone remodeling in the lesions confirmed by radiography suggested that osteonecrosis was not the cause, but the result of infection.     

Central giant cell granuloma: treatment with calcitonin, triamcinolone acetonide, and a cystic finding 3 years and 6 months after the primary treatment

Background

Central giant cell granuloma is a benign condition of the jaws which may present an aggressive behavior.

Case report

A 9-year-old boy was complaining of swelling in the floor of the mouth. A solid swelling was observed in the area of the lower incisors. From the radiographic exam, we observed a radiolucent image in the mandibular bone with well-defined limits extending from the apical region of tooth 33 to the apical region of tooth 42.

Discussion

Due to the diagnosis and the age of the patient, we chose a conservative treatment, administering subcutaneous injections of calcitonin. During this treatment, no reduction to the lesion was observed. Therefore, we chose to treat the lesion with triamcinolone acetonide. Monthly follow-ups demonstrated good lesion reduction and the absence of any clinical symptoms during the first 2 years. After a 3-year follow-up, the patient returned, presenting mobility of the lower incisors. A significant increase in the size of the lesion was observed. After a biopsy, with the removal of tissue which had the appearance of a cyst capsule, microscopic analyses were found to be compatible with a secondarily infected cyst. Two months following this procedure, the patient did not present tooth mobility anymore and the oral mucosa presented a normal aspect. Following a radiographic exam, full lesion repair was observed. These conservative treatments should be the first option in cases of central giant cell granuloma and the patient must be observed for a long period of time, until no further clinical or radiographic signs of lesions are observed.     

Unusual form of recurrent giant cell granuloma of the mandible and lower extremities in a patient with neurofibromatosis type 1

We report on a girl with familial neurofibromatosis type 1 (Nf1) who at age 11 had multiple osteolytic lesions of the right mandible and the distal femoral and proximal tibial metaphyses bilaterally. No other skeletal component was affected, and no abnormalities were present on skeletal radiographs of her parents. Histologic examination of the mandibular and right femoral lesions revealed a “multinucleated giant cell process.” Results of repeated routine laboratory and bone metabolic studies were within normal limits. The lack of reliable histologic criteria and the diagnostic problems in distinguishing among central giant cell granulomata, giant cell tumors of the jaw, cherubism, brown tumors resulting from (occult) hyperparathyroidism, Jaffé-Campanacci syndrome, McCune-Albright syndrome, Noonan-like/multiple giant cell lesion syndrome, and multiple nonossifying fibromas of bone are discussed. (Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1999;87:67-72)     

Presence of cholesterol granuloma in the maxillary and ethmoid sinuses

Introduction

Although cholesterol granuloma associated with chronic middle ear disease is shown to be common in the mastoid antrum and air cells of the temporal bone (Leon et al., Arch Pathol Lab Med 126:217–219, 2002), its presence in the maxillary and ethmoid sinuses is rarely encountered. There are few cases reported regarding the incidence of cholesterol granuloma in these sinuses (Ko et al., Am J Otoryngol 27:370–372, 2006).

Case

Here, we report a case of concomitant cholesterol granuloma in the maxillary and ethmoid sinuses of a 33-year-old man who underwent surgical excision.

Discussion

Histopathological examination of the removed specimen revealed fragments of respiratory mucosa with cholesterol clefts surrounded by multinucleated foreign-body giant cells.     

Inflammatory myofibroblastic tumour of the maxillary sinus and the oral cavity

Background

Inflammatory myofibroblastic tumours (IMT) are benign lesions that mimic malignant neoplastic processes due to their infiltrating and destructive nature. They can be found anywhere in the body, although they are most commonly located in the lung. IMTs of the head and neck region are rare entities.

Case report

We report a case of a maxillary IMT with an enlarged intraoral component which was adequately treated with surgical excision without long-lasting additional corticosteroid treatment.

Discussion

Inflammatory myofibroblastic tumours can be easily misdiagnosed as malignant neoplastic processes due to their destructive nature. Proper diagnosis is essential to avoid mutilating and disfiguring surgical procedures.     

Correlation between hand/wrist and panoramic radiographs in severe secondary hyperparathyroidism

Objectives

Hand/wrist and dental radiographs are important for osteoporosis analysis in secondary hyperparathyroidism (SHPT). This study evaluated whether a correlation exists between the effects of the disease on the hands and jaws, and investigated the association between osteoporosis progression in the hands and parathyroid hormone (PTH) levels in chronic kidney disease (CKD) patients.

Materials and methods

Four panoramic radiographic parameters (mental index, mandibular cortical index, trabecular bone pattern, and calcification/resorption) and four corresponding hand/wrist radiographic parameters (metacarpal cortical thickness, phalangeal cortical index, trabecular bone pattern, and calcification/resorption) were applied to investigate possible correlation between the effects of SHPT on the jaws and hands/wrists, by Spearman's correlation coefficient. PTH levels and the hand/wrist radiographic parameters were also tested by spearman's correlation coefficient (p?<?0.05). The presence of brown tumors, vascular calcifications, and acroosteolysis on the hands was also evaluated.

Results

Mandibular cortical index was strongly correlated with the phalangeal cortical index (p?=?0.000). Phalangeal cortical index and trabecular bone pattern of hand/wrist correlated with PTH levels (0.002 and 0.000, respectively). Brown tumors occurred in four CKD patients, while both vascular calcifications and acroosteolysis were observed in 19 patients.

Conclusion

There is a significant correlation between the morphological changes caused by secondary hyperparathyroidism in hand and jaw bones. The morphological status can be assessed using the mandibular cortical index, besides the phalangeal cortical index. The latter correlates well with parathyroid hormone levels of advanced chronic kidney disease.

Clinical relevance

Panoramic images reveal morphological changes in the jaw bone, indicating likewise changes in the hand/wrist in severe secondary hyperparathyroidism. The severity of the bone changes may be a reflection of the parathyroid hormone levels in advanced chronic kidney disease.     

8-Year Follow-up of Central Giant Cell Lesion Mimicking Apical Periodontitis

Introduction

Lesions of nonendodontic origin may mimic apical periodontitis. Central giant cell lesions (CGCLs) are aggressive or nonaggressive benign idiopathic intraosseous lesions of the jaw. This report describes a case of a CGCL in the periapical region of teeth #21–#26 of a 17-year-old female who sought orthodontic care because of a change in the position of tooth #23.

Methods

Clinical examination revealed mild facial asymmetry caused by increased volume in the mental region and cortical bone expansion but no cortical disruption. A panoramic radiograph showed a well-defined radiolucent osteolytic lesion involving teeth #21–#26. The cortical bone was not affected, and there was no root resorption. Incisional biopsy was performed, and the diagnosis was a CGCL. The lesion was enucleated surgically. CGCLs should be included in the differential diagnosis of jaw lesions that mimic apical periodontitis.

Results

The patient subsequently underwent orthodontic treatment successfully.

Conclusions

The 8-year clinical and radiographic follow-up confirmed lesion remission, no recurrence, and pulp vitality of all teeth.     

Condylar metastasis from prostatic carcinoma mimicking temporomandibular disorder: a case report

Background

Although metastatic carcinoma is the most common malignant tumor of the bone, less than 1% of all metastatic bone lesions are presented in the maxillofacial area. As the mandibular body is the most frequent localization, metastasis to the mandibular condyle is extremely rare.

Case report

This report describes a rare case of prostate carcinoma metastatic to the mandibular condyle in a 75-year old man, who was referred because of persistent pain in the temporomandibular joint (TMJ) region and a limitation of opening, initially misdiagnosed and treated as temporomandibular disorder (TMD). Histopathological examination confirmed the suspected metastasis of prostate carcinoma and local radiation therapy was performed.

Discussion

TMD represent a diagnostic challenge and sometimes an interdisciplinary approach is required to prevent a delay of the correct treatment. Metastatic cancer should be included in the differential diagnosis of TMD, especially in patients with a malignant disease.     

Traumatic bone cyst, aneurysmal bone cyst, and central giant cell granuloma--pathogenetically related lesions?

Three central bone lesions, a traumatic bone cyst, an aneurysmal bone cyst, and a central giant cell granuloma are reported. Comparisons of the clinical, radiographic, and histological features were made. Although these lesions are recognized as separate and distinct pathological entities, a common pathogenesis is suggested by similarities in certain features. Diagnostic aids are suggested to assist in a differential diagnosis.     

A histological comparison of the giant cells in the central giant cell granuloma of the jaws and the giant cell tumor of long bone

Abstract. A histological comparison has been made of the giant cells in 10 cases which had been diagnosed as central giant cell granuloma of the jaws and 10 cases diagnosed as giant cell tumor of long bone. The numbers of nuclei in each of 50 giant cells were counted in the 20 lesions. The giant cells were measured in order to compare their sizes in the respective lesions. It was found that the giant cells of the jaw lesions contained significantly fewer nuclei than those of the lesions in other bones. Secondly it was found that cells in the lesions of long bone have significantly larger dimensions than those in the jaw lesions. There was, however, an area of overlap. It was concluded that these data supported the view that giant cell tumor and giant cell granuloma are distinct entities. Some giant cell lesions of long bones are, however, morphologically indistinguishable from giant cell lesions of the jaws; and conversely giant cell lesions of the jaws are indistinguishable from some giant cell lesions of the long bones. It seems possible, therefore, that some jaw lesions are giant cell tumors and that some giant cell lesions outside the jaws are giant cell granulomas.     

A histological comparison of the giant cells in the central giant cell granuloma of the jaws and the giant cell tumor of long bone

A histological comparison has been made of the giant cells in 10 cases which had been diagnosed as central giant cell granuloma of the jaws and 10 cases diagnosed as giant cell tumor of long bone. The numbers of nuclei in each of 50 giant cells were counted in the 20 lesions. The giant cells were measured in order to compare their sizes in the respective lesions. It was found that the giant cells of the jaw lesions contained significantly fewer nuclei than those of the lesions in other bones. Secondly it was found that cells in the lesions of long bone have significantly larger dimensions than those in the jaw lesions. There was, however, an area of overlap. It was concluded that these data supported the view that giant cell tumor and giant cell granuloma are distinct entities. Some giant cell lesions of long bones are, however, morphologically indistinguishable from lesions of the jaws; and conversely giant cell lesions of the jaws are indistinguishable from some giant cell lesions of the long bones. It seems possible, therefore, that some jaw lesions are giant cell tumors and that some giant cell lesions outside the jaws are giant cell granulomas.     

Multifocal central giant cell lesions of the maxillofacial skeleton: a case report

The classification of giant cell lesions of the maxillofacial skeleton is one that remains controversial. Classifying giant cell lesions of the jaw as granulomatous based solely on location seems inappropriate. The categories of benign or malignant are more realistic. Benign lesions may then be subdivided into aggressive and nonaggressive. Multifocal giant cell lesions strongly suggest the brown tumor of hyperparathyroidism. Serum chemistry tests including calcium, phosphorus, ionized calcium, and PTH levels should routinely be obtained when a giant cell lesion is suspected. A case of benign, aggressive, multifocal central giant cell lesions of the maxillofacial skeleton, in the absence of either primary or secondary hyperparathyroidism is presented. Whether this represents metastasis from the initial lesion, metabolic osteoclastic dysfunction, or a new entity, craniofacial giant cell dysplasia, is unknown.     

Role of FNAC in the diagnosis of intraosseous jaw lesions

Background

FNAC of intraosseous jaw lesions has not been widely utilized for diagnosis due to rarity and diversity of these lesions, limited experience and lack of well established cytological features. Aim of the study was to determine the role of FNAC in the diagnosis of intraosseous jaw swellings.

Material and Methods

42 patients underwent FNAC over a period of 7 years (2007-2013), of which 37 (88.1%) aspirates were diagnostic. Histopathology correlation was available in 33 cases and diagnostic accuracy of FNAC was calculated.

Results

Lesions were categorized into inflammatory 3, cysts/hamartomas 15 and neoplasms 19. Mandibular and maxillary involvement was seen in 21 and 16 patients respectively. Of these, benign cysts and malignant lesions were commonest, accounting for 27% lesions (10 cases) each. One case of cystic ameloblastoma was misdiagnosed as odontogenic cyst on cytology. Overall, sensitivity and specificity of FNAC were 94.7% and 100% respectively with a diagnostic accuracy of 97.3%. Definitive categorization of giant cell lesions, fibro-osseous lesions, odontogenic tumors and cystic lesions was not feasible on FNAC.

Conclusions

FNAC is a simple, safe and minimally invasive first line investigation which can render an accurate preoperative diagnosis of intraosseous jaw lesions, especially the malignant ones in the light of clinic-radiological correlation. Key words: Jaw swellings, intraosseous, FNAC.