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"Journal of Clinical and Diagnostic Research is at present a well-known Indian originated scientific journal which started with a humble beginning. I have been associated with this journal since many years. I appreciate the Editor, Dr. Hemant Jain, for his constant effort in bringing up this journal to the present status right from the scratch. The journal is multidisciplinary. It encourages in publishing the scientific articles from postgraduates and also the beginners who start their career. At the same time the journal also caters for the high quality articles from specialty and super-specialty researchers. Hence it provides a platform for the scientist and researchers to publish. The other aspect of it is, the readers get the information regarding the most recent developments in science which can be used for teaching, research, treating patients and to some extent take preventive measures against certain diseases. The journal is contributing immensely to the society at national and international level."



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Dr. Arundhathi. S
MBBS, MD (Pathology),
Sanjay Gandhi institute of trauma and orthopedics,
Bengaluru.
On Aug 2018




Dr. Mamta Gupta,
"It gives me great pleasure to be associated with JCDR, since last 2-3 years. Since then I have authored, co-authored and reviewed about 25 articles in JCDR. I thank JCDR for giving me an opportunity to improve my own skills as an author and a reviewer.
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Dr. Mamta Gupta
Consultant
(Ex HOD Obs &Gynae, Hindu Rao Hospital and associated NDMC Medical College, Delhi)
Aug 2018




Dr. Rajendra Kumar Ghritlaharey

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Thanking you
With sincere regards
Dr. Rajendra Kumar Ghritlaharey, M.S., M. Ch., FAIS
Associate Professor,
Department of Paediatric Surgery, Gandhi Medical College & Associated
Kamla Nehru & Hamidia Hospitals Bhopal, Madhya Pradesh 462 001 (India)
E-mail: drrajendrak1@rediffmail.com
On May 11,2011




Dr. Shankar P.R.

"On looking back through my Gmail archives after being requested by the journal to write a short editorial about my experiences of publishing with the Journal of Clinical and Diagnostic Research (JCDR), I came across an e-mail from Dr. Hemant Jain, Editor, in March 2007, which introduced the new electronic journal. The main features of the journal which were outlined in the e-mail were extensive author support, cash rewards, the peer review process, and other salient features of the journal.
Over a span of over four years, we (I and my colleagues) have published around 25 articles in the journal. In this editorial, I plan to briefly discuss my experiences of publishing with JCDR and the strengths of the journal and to finally address the areas for improvement.
My experiences of publishing with JCDR: Overall, my experiences of publishing withJCDR have been positive. The best point about the journal is that it responds to queries from the author. This may seem to be simple and not too much to ask for, but unfortunately, many journals in the subcontinent and from many developing countries do not respond or they respond with a long delay to the queries from the authors 1. The reasons could be many, including lack of optimal secretarial and other support. Another problem with many journals is the slowness of the review process. Editorial processing and peer review can take anywhere between a year to two years with some journals. Also, some journals do not keep the contributors informed about the progress of the review process. Due to the long review process, the articles can lose their relevance and topicality. A major benefit with JCDR is the timeliness and promptness of its response. In Dr Jain's e-mail which was sent to me in 2007, before the introduction of the Pre-publishing system, he had stated that he had received my submission and that he would get back to me within seven days and he did!
Most of the manuscripts are published within 3 to 4 months of their submission if they are found to be suitable after the review process. JCDR is published bimonthly and the accepted articles were usually published in the next issue. Recently, due to the increased volume of the submissions, the review process has become slower and it ?? Section can take from 4 to 6 months for the articles to be reviewed. The journal has an extensive author support system and it has recently introduced a paid expedited review process. The journal also mentions the average time for processing the manuscript under different submission systems - regular submission and expedited review.
Strengths of the journal: The journal has an online first facility in which the accepted manuscripts may be published on the website before being included in a regular issue of the journal. This cuts down the time between their acceptance and the publication. The journal is indexed in many databases, though not in PubMed. The editorial board should now take steps to index the journal in PubMed. The journal has a system of notifying readers through e-mail when a new issue is released. Also, the articles are available in both the HTML and the PDF formats. I especially like the new and colorful page format of the journal. Also, the access statistics of the articles are available. The prepublication and the manuscript tracking system are also helpful for the authors.
Areas for improvement: In certain cases, I felt that the peer review process of the manuscripts was not up to international standards and that it should be strengthened. Also, the number of manuscripts in an issue is high and it may be difficult for readers to go through all of them. The journal can consider tightening of the peer review process and increasing the quality standards for the acceptance of the manuscripts. I faced occasional problems with the online manuscript submission (Pre-publishing) system, which have to be addressed.
Overall, the publishing process with JCDR has been smooth, quick and relatively hassle free and I can recommend other authors to consider the journal as an outlet for their work."



Dr. P. Ravi Shankar
KIST Medical College, P.O. Box 14142, Kathmandu, Nepal.
E-mail: ravi.dr.shankar@gmail.com
On April 2011
Anuradha

Dear team JCDR, I would like to thank you for the very professional and polite service provided by everyone at JCDR. While i have been in the field of writing and editing for sometime, this has been my first attempt in publishing a scientific paper.Thank you for hand-holding me through the process.


Dr. Anuradha
E-mail: anuradha2nittur@gmail.com
On Jan 2020

Important Notice

Case report
Year : 2023 | Month : December | Volume : 17 | Issue : 12 | Page : ZD12 - ZD16 Full Version

Aneurysmal Bone Cyst with Ossifying Fibroma of the Mandible: A Case Report and Review of the Literature


Published: December 1, 2023 | DOI: https://doi.org/10.7860/JCDR/2023/66737.18835
Samiha Jameel Ahmed Khan, Madhuri Gawande, Alka Hande, Swati Patil, Archana Sonone

1. Postgraduate Student, Department of Oral Pathology and Microbiology, Sharad Pawar Dental College and Hospital, Datta Meghe Institute of Higher Education and Research, Sawangi, Wardha, Maharashtra, India. 2. Professor and Head, Department of Oral Pathology and Microbiology, Sharad Pawar Dental College and Hospital, Datta Meghe Institute of Higher Education and Research, Sawangi, Wardha, Maharashtra, India. 3. Professor, Department of Oral Pathology and Microbiology, Sharad Pawar Dental College and Hospital, Datta Meghe Institute of Higher Education and Research, Sawangi, Wardha, Maharashtra, India. 4. Associate Professor, Department of Oral Pathology and Microbiology, Sharad Pawar Dental College and Hospital, Datta Meghe Institute of Higher Education and Research, Sawangi, Wardha, Maharashtra, India. 5. Assistant Professor, Department of Oral Pathology and Microbiology, Sharad Pawar Dental College and Hospital, Datta Meghe Institute of Higher Education and Research, Sawangi, Ward

Correspondence Address :
Samiha Jameel Ahmed Khan,
Postgraduate Student, Department of Oral Pathology and Microbiology, Sharad Pawar Dental College and Hospital, Datta Meghe Institute of Higher Education and Research, Sawangi, Wardha-442001, Maharashtra, India.
E-mail: samiha.khan26@gmail.com

Abstract

Aneurysmal Bone Cysts (ABCs) are uncommon benign bone lesions primarily affecting children and adolescents. They are distinguished by severe bone breakdown and expansive tissue growth, resulting in clinical symptoms and potential complications. ABCs typically occur in long bones, but reports have also documented their presence in the jaws, particularly the mandible. These lesions are commonly considered non-cancerous and are characterised by cystic or blood-filled chambers. There are two clinicopathological variations of ABC: primary ABC and secondary ABC. Primary ABC originates independently, while secondary ABC develops as a result of a pre-existing lesion such as a cyst, tumor, or Fibro-Osseous Lesions (FOL) like solitary bone cyst, ossifying fibroma, or giant cell granuloma. When ABC coexists with another bone lesion believed to be its precursor, it is referred to as an “ABC plus lesion”. In this case report, a 75-year-old patient, presented with a painful growth in the lower anterior region of the jaw for the past 18 months. The diagnosis was established through clinical, radiological, and histopathological examinations. A complete surgical resection was performed, followed by uneventful reconstruction. Histopathological examination confirmed the presence of ABC with ossifying fibroma (ABC plus lesion). It is important to address ABC plus lesions as they can cause significant pain, deformity, and discomfort. Although non-cancerous, they can still disrupt normal bone structure and function. This case report emphasises the clinical, radiographic, and histopathological features of ABC plus lesions, aiding in disease identification.

Keywords

Benign, Bone breakdown, Odontogenic, Resection, Reconstruction

Case Report

A 75-year-old female patient reported to the outpatient department with a growth over the lower front region of the jaw that had been present for approximately 18 months. Initially small, the growth had rapidly increased in size. The patient tested positive for Hepatitis B surface Antigen (HBsAg) and had a history of hypertension.

During the extraoral examination, a diffuse growth was observed over the anterior region of the mandible, extending anteroposteriorly from the left corner of the mouth to the right corner of the mouth, and superoinferiorly from the vermillion border of the upper lip to 3 cm above the inferior border of the mandible. The growth measured approximately 7.5×6 cm and had a firm consistency. It was non-tender (Table/Fig 1).

Intraoral examination revealed insufficient mouth opening. A 7×5 cm lesion was observed, extending mesiodistally from the 45 to 35 region and superoinferiorly from the gingival border of the lower anterior teeth into the lingual and gingivolabial sulcus. The edges were smooth with well-defined borders, and the consistency ranged from soft to firm (Table/Fig 2).

Radiographic examinations were performed. The Orthopantomogram (OPG) revealed a well-defined multilocular radiolucency in the mandibular symphysis region, extending from the 35 to 46 region (Table/Fig 3). The Computed Tomography (CT) scan (Table/Fig 4) showed a 7.5×5.3×6.2 cm expansile large cystic lesion in the mandibular symphysis menti. A provisional diagnosis of Central Giant Cell Granuloma (CGCG) was made. The differential diagnosis at the time included ameloblastoma and keratocystic odontogenic tumour, also known as Odontogenic Keratocyst (OKC).

Under general anaesthesia, a segmental mandibulectomy was performed from the angle of the mandible (right) to the 38 region on the left side of the mandible. Reconstruction was carried out using a Pectoralis Major Myocutaneous Flap (PMMC) on the right side, and a tracheostomy was performed. The resected specimen was sent for histopathological examination (Table/Fig 5).

During gross examination, an exophytic greyish-black extensive lesion measuring 8×7×6 cm was observed on the lower anterior jaw (Table/Fig 6). On the cut section, excessive bleeding was encountered, resembling a sponge soaked in blood with cavernous spaces (Table/Fig 7).

Histopathological examination of the Haematoxylin and Eosin (H&E) stained lesional tissue section showed a fibrocellular connective tissue stroma with large, cavernous or sinusoidal spaces filled with blood (Table/Fig 8). The connective tissue stroma exhibited multi-nucleated giant cells with 10-15 nuclei and immature plump fibroblasts (Table/Fig 9). Other sections revealed immature bony trabeculae lined by osteoblastic rimming and numerous osteocytes (Table/Fig 10). The connective tissue stroma exhibited delicate interlacing collagen fibrils interspersed with a large number of actively proliferating fibroblasts (Table/Fig 11). A final diagnosis of aneurysmal bone cyst with ossifying fibroma was made. No special stains were used in this case.

The patient experienced full recovery and had no pain or other symptoms after six months of follow-up. There was no evidence of recurrence (Table/Fig 12),(Table/Fig 13).

Discussion

ABC is a rare non-cancerous osteolytic tumour of bone tissue characterised by multiple sponge-like, blood-containing areas of varying sizes. These areas often lack endothelial lining and contain bone elements and osteoclast-like giant cells (1),(2). ABCs are commonly seen in areas with greater marrow and venous content. Due to the low venous pressure in skull bones, ABCs can be unpredictable lesions (3). They are usually found in the metaphysis of long bones, such as the tibia and femur (more than 50%), and in the spine (12-30%) (4). ABCs are less common in the craniofacial 14skeleton (2-12%) (4),(5),(6). However, the mandible is a common location for ABCs in the head-neck region (6).

ABC is divided into three categories. The traditional or vascular type presents as an expansile, rapidly expanding destructive lesion that causes cortical perforation and soft tissue invasion. The solid variant may appear as a small, painless growth initially detected as a radiolucent lesion on routine radiographs, or as a clinically significant tumour (7),(8). The mixed type exhibits characteristics of both the vascular (classic) and solid varieties. Rapid expansion and activation of stable lesions have been documented, suggesting that it may represent a transient phase of the lesion (8).

Clinicopathologically, ABC has two forms: primary (congenital or acquired) and secondary, which arises from a pre-existing lesion. Congenital ABC is determined by factors such as arteriovenous malformation, tooth growth, and tooth maturation during infancy. Acquired ABC is often associated with trauma. Secondary ABC can be linked to the progression of pre-existing lesions, such as a cyst, tumour, or fibro-osseous lesions like solitary bone cysts, ossifying fibromas, or giant cell granulomas (9).

Ossifying Fibroma (OF), a benign bone tumour usually referred to as a form of FOL, can affect both the mandible and maxilla, but it is more frequently observed in the mandible, accounting for 70-90% of cases (10). Clinically, this tumour manifests as a slowly expanding intrabony tumour that often lacks symptoms and rarely grows to a size that causes facial asymmetry (11). Only a few cases in the literature describe the close association between ossifying fibroma and ABCs, as in the present case (12). In the facial bones, the occurrence of ABCs subsequent to ossifying fibroma is still a rare finding (13).

A literature review of thirty-two ABC-plus-lesions revealed that males are more commonly affected, and there is a higher propensity for mandibular involvement. Sixty-eight percent of the thirty-two ABC-plus-lesions were associated with FOLs, while giant cell lesions accounted for 32% of the cases. ABC presents with a wide range of clinical characteristics, ranging from a painless lesion identified through radiographic examination to a symptomatic (painful), expanding, and destructive pattern (14). However, the majority of cases were painless, as was the situation in this case. In contrast to ABCs in long bones, ABC-plus-lesions typically cause discomfort and exhibit a tendency for rapid growth, whereas ABCs in other locations are often associated with malignant tumours such as osteosarcomas and chondrosarcomas (15).

The radiological appearance of jaw ABCs is highly variable. The lesion may exhibit bony growth, a cyst-like appearance resembling a soap bubble or honeycomb, or it may have an unconventional inflated appearance. The cortex may be perforated or destroyed, and a periosteal response may occur (16). The lesion can appear as radiopaque, radiolucent, or mixed. In this case, a multiocular radiolucency was observed, causing expansion of the cortical plates and thinning of the inferior mandibular border. Root resorption in the affected teeth was also noted. However, the diagnosis based solely on radiographic examination is uncertain, as other lesions, such as ameloblastoma, odontogenic cysts or tumours, myxoma, or central haemangiomas, can have similar radiographic appearances (17).

ABC exhibits numerous sinusoidal spaces filled with blood in a fibrocellular connective tissue stroma, along with bone/osteoid material and multiple multinucleated giant cells. The presence of haemosiderin pigments is also variably observed, which are pathognomonic features of the vascular variant of ABC (18). On the other hand, the solid variant shows foci of haemorrhage with numerous fibroblasts and fibrohistiocytes. Additionally, osteoclast-like giant cells, areas of osteoblastic differentiation with bone elements, and calcifying fibromyxoid tissue are present. The mixed variant demonstrates both solid and vascular characteristics. Aneurysmal bone cyst plus-lesions exhibit a combination of classic (vascular) and solid forms, along with related lesions, featuring multiple vascular spaces in the fibrocellular stroma, multinucleated giant cells, and bone tissue production (19). The histological findings in this case were consistent with the aforementioned criteria, indicating an ABC with OF.

The pathophysiology of ABCs is still a subject of debate. It could be caused by reactive vascular malformation, post-traumatic stress disorder, or hereditary susceptibility (20).

In most cases, total resection of the lesion is the preferred treatment modality for ABC plus-lesions. Surgical resection and curettage are considered the gold standard treatment. Diagnostic and therapeutic embolisation, curettage, block resection, radiation reconstruction, and systemic therapy with calcitonin are also utilised. Some studies have reported self-healing over a long follow-up period. For patients with aesthetic deformity, mandible discontinuity, or a high risk of fracture, early restoration of the defect using autogenous grafts has been recommended (5),(14),(21),(22),(23).

In this case, surgical resection was performed as the treatment approach, and regular monitoring was conducted. No evidence of residual lesion was observed after six months of follow-up. The case reports of ABC plus-lesions in the head and neck region have been compiled in (Table/Fig 14) (3),(24),(25),(26),(27),(28),(29),(30),(31),(32),(33),(34),(35),(36),(37),(38),(39),(40),(41).

Conclusion

The ABC is often associated with fibro-osseous jaw lesions, which are referred to as aneurysmal bone cyst plus-lesions. The clinical, radiological, and histological data were all considered in this case of a secondary ABC-plus-lesion. Due to the diverse pathophysiology of ABC-plus lesions, diagnosing and identifying them, as well as determining the appropriate therapy, poses a challenge for surgeons. A definitive diagnosis can only be obtained through an incisional biopsy. Biopsies should be performed after ruling out vascular lesions.

References

1.
Rosenberg AE, Nielsen GP, Fletcher JA. Aneurysmal bone cyst. In: Fletcher CD, Unni KK, Mertens F, editors. WHO Classification of Tumors: Pathology and Genetics of Tumors of Soft Tissue and Bone. 3rd ed. Lyon: IARC Press; 2005. Pp. 338-39.
2.
Jundt G. Aneurysmal bone cyst. In: Barnes L, Eveson JW, Reichart P, Sidransky D, editors. WHO Classification of Tumors: Pathology and Genetics of Head and Neck Tumors. 3rd ed. Lyon: IARC Press; 2005. Pp. 326.
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Moghe S, Saini N, Pillai A, Moghe A, Pillai K. Aneurysmal bone cyst plus in an 8-year-old female, a case report. IOSR J Dent Med Sci. 2014;13:63-68. Doi: 10.9790/0853-13466368. [crossref]
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Perrotti V, Rubini C, Fioroni M, Piattelli A. Solid aneurysmal bone cyst of the mandible. Int J Pediatr Otorhinolaryngol. 2004;68(10):1339-44. [crossref][PubMed]
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Rapidis AD, Vallianatou D, Apostolidis C, Lagogiannis G. Large lytic lesion of the ascending ramus, the condyle, and the infratemporal region. J Oral Maxillofac Surg. 2004;62(8):996-1001. [crossref][PubMed]
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Bataineh AB. Aneurysmal bone cysts of the maxilla: A clinicopathologic review. J Oral Maxillofac Surg. 1997;55(11):1212-16. [crossref][PubMed]
7.
López-Arcas JM, Cebrián L, González J, Burgueño M. Aneurysmal bone cyst of the mandible: Case presentation and review of the literatura. Med Oral Patol Oral Cir Bucal. 2007;12:E401-03. https://scielo.isciii.es/pdf/medicorpa/v12n5/15.pdf.
8.
Pelo S, Gasparini G, Boniello R, Moro A, Amoroso PF. Aneurysmal bone cyst is located in the mandibular condyle. Head Face Med. 2009;5:8. Doi: 10.1186/1746-160X-5-8. [crossref][PubMed]
9.
Svensson B, Isacsson G. Benign osteoblastoma associated with an aneurysmal bone cyst of the mandibular ramus and condyle. Oral Surg Oral Med Oral Pathol. 1993;76:433-36. Doi: 10.1016/0030-4220(93)90008-r. [crossref][PubMed]
10.
Liu Y, Wang H, You M, Yang Z, Miao J, Shimizutani K, et al. Ossifying fibromas of the jaw bone: 20 cases. Dentomaxillofacial Radiology. 2010;39(1):57-63. [crossref][PubMed]
11.
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DOI and Others

DOI: 10.7860/JCDR/2023/66737.18835

Date of Submission: Jul 26, 2023
Date of Peer Review: Sep 11, 2023
Date of Acceptance: Oct 11, 2023
Date of Publishing: Dec 01, 2023

AUTHOR DECLARATION:
• Financial or Other Competing Interests: None
• Was informed consent obtained from the subjects involved in the study? Yes
• For any images presented appropriate consent has been obtained from the subjects. Yes

PLAGIARISM CHECKING METHODS:
• Plagiarism X-checker: Jul 27, 2023
• Manual Googling: Sep 19, 2023
• iThenticate Software: Oct 09, 2023 (11%)

ETYMOLOGY: Author Origin

EMENDATIONS: 6

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