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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.
It 's a multispecialty journal, publishing high quality articles. It gives a platform to the authors to publish their research work which can be available for everyone across the globe to read. The best thing about JCDR is that the full articles of all medical specialties are available as pdf/html for reading free of cost or without institutional subscription, which is not there for other journals. For those who have problem in writing manuscript or do statistical work, JCDR comes for their rescue.
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Reviewing articles is no less a pain staking process and requires in depth perception, knowledge about the topic for review. It requires time and concentration, yet I enjoy doing it. The JCDR website especially for the reviewers is quite user friendly. My suggestions for improving the journal is, more strict review process, so that only high quality articles are published. I find a a good number of articles in Obst. Gynae, hence, a new journal for this specialty titled JCDR-OG can be started. May be a bimonthly or quarterly publication to begin with. Only selected articles should find a place in it.
An yearly reward for the best article authored can also incentivize the authors. Though the process of finding the best article will be not be very easy. I do not know how reviewing process can be improved. If an article is being reviewed by two reviewers, then opinion of one can be communicated to the other or the final opinion of the editor can be communicated to the reviewer if requested for. This will help one’s reviewing skills.
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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

"I wish to thank Dr. Hemant Jain, Editor-in-Chief Journal of Clinical and Diagnostic Research (JCDR), for asking me to write up few words.
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In the era of fast growing newer technologies, and in computer and internet friendly environment the manuscripts preparation, submission, review, revision, etc and all can be done and checked with a click from all corer of the world, at any time. Of course there is always a scope for improvement in every field and none is perfect. To progress, one needs to identify the areas of one's weakness and to strengthen them.
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Every one of us: authors, reviewers, editors, and publisher are responsible for enhancing the stature of the journal. I wish for a great success for JCDR."



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 : 2024 | Month : March | Volume : 18 | Issue : 3 | Page : ZD08 - ZD11 Full Version

Cystic Extrafollicular Adenomatoid Odontogenic Tumour: A Case Report and Pictorial Update


Published: March 1, 2024 | DOI: https://doi.org/10.7860/JCDR/2024/68667.19119
Deepak Pandiar, P Anbumani, Reshma Poothakulath Krishnan, S Dharini

1. Associate Professor, Department of Oral Pathology and Microbiology, Saveetha Dental College and Hospitals, Saveetha Institute of Medical and Technical Sciences, Saveetha University, Chennai, Tamil Nadu, India. 2. General Practitioner (Oral and Maxillofacial Surgeon), 115, Vellam Thangiya Pillayar Kovil Street, Tirunelveli Town, Tamil Nadu, India. 3. Assistant Professor, Department of Oral Pathology and Microbiology, Saveetha Dental College and Hospitals, Saveetha Institute of Medical and Technical Sciences, Saveetha University, Chennai, Tamil Nadu, India. 4. Postgraduate Resident, Department of Oral Pathology and Microbiology, Saveetha Dental College and Hospitals, Saveetha Institute of Medical and Technical Sciences, Saveetha University, Chennai, Tamil Nadu, India.

Correspondence Address :
Dr. Deepak Pandiar,
Associate Professor, Department of Oral Pathology and Microbiology, Saveetha Dental College and Hospitals, Saveetha Institute of Medical and Technical Sciences, Saveetha University, Chennai-600077, Tamil Nadu, India.
E-mail: deepakpandiar1923@yahoo.com

Abstract

Adenomatoid Odontogenic Tumour (AOT) constitutes about 5% of all odontogenic tumours and is most commonly seen in young females, in association with impacted maxillary canines in a Dentigerous Cyst (DC) like relationship. Extrafollicular AOT is an uncommon variant seen unassociated with impacted teeth. Rare cystic presentation is reported with most cases seen arising from or in association with DC. Here, the authors present a case of extrafollicular AOT in a 29-year-old Dravidian male, who reported with a chief complaint of swelling of lower anterior jaw region. Radiographically, a well-defined radiolucent lesion was noted extending from mesial aspect of the roots of right mandibular second molar, crossing the midline to involve the body of mandible till mesial aspect of lower left canine. There was evidence of cortical perforation. The lesion was excised and sent for histopathological evaluation. Histopathologically, the lesion was diagnosed as cystic AOT-extrafollicular variant. There was adequate healing and no signs of recurrence or residual disease were noted 18 months after the surgery. The present case is a unique presentation of cystic extrafollicular AOT with pictorial demonstration and detailed explanation of its pathogenesis.

Keywords

Dentigerous cyst, Orthopantomogram, Protein

Case Report

A 29-year-old Dravid Indian male reported with a chief complaint of swelling of lower anterior jaw region for one and a half years. The swelling was slow in onset with no associated pain or pus discharge. On intraoral examination, an ill-defined bony hard swelling was noted centered to the right anterior body of the mandible crossing the midline with buccal and lingual cortical expansion and obliteration of buccal sulcus. The swelling was non-tender, non-fluctuant, non-pulsatile and non-compressible with no associated bleeding or pus discharge. All the teeth were vital. The Orthopantomogram (OPG) showed a well-defined radiolucent lesion extending from mesial aspect of the roots of 47, crossing the midline to involve the body of the mandible till mesial aspect of 33 (Table/Fig 1)a. There was buccal and cortical plate perforation and concomitant root resorption. The findings were confirmed by Computed Tomography (CT) (Table/Fig 1)b. The lesion was provisionally diagnosed as developmental odontogenic cyst, however considering the bony expansion and root resorption the existence of odontogenic neoplasm could not be completely ruled out. Two mL of straw-coloured watery fluid was aspirated (Table/Fig 1)c; however, the cytological features were non contributory to the diagnosis. A total protein of 6.7 g per 100 mL was estimated. As the protein content was >4 g/100 mL, a non keratinising cystic lesion was considered and the lesion was thus enucleated under local anaesthesia and sent for histopathological examination (Table/Fig 1)d,(Table/Fig 2).

Histopathological examination showed tissue which predominantly appeared cystic in architecture. The cystic lining was stratified squamous and lacked the classical features of any of the named odontogenic cysts and tumours such as Dentigerous Cyst (DC), Odontogenic Keratocyst (OKC), Calcifying Odontogenic Cyst (COC), Glandular Odontogenic Cyst (GOC) or unicystic ameloblastoma. The lining was predominantly non-keratinised, 4-6 layered thick with superficial loosening of cells and heaped up appearance of surface cells (Table/Fig 3)a-d. There was evidence of proliferation of thin strands and bud like extensions from the lining into the wall (Table/Fig 3)e,f. Basophilic calcifications were noted close to the lining (Table/Fig 3)b. At areas, the cyst wall showed hyalinisation/inductive changes particularly sub-epithelially and around the strands (Table/Fig 4)a,b. The other areas consisted of odontogenic epithelial cells arranged in plexus and solid nodules (Table/Fig 4)c,d. The cells were cuboidal/spindle/polygonal in shape with scanty eosinophilic to clear cytoplasm and deeply basophilic monomorphous round to oval nuclei. At areas, the tumour cells were arranged in pseudo rosettes and double convoluted structures with hyaline ring (Table/Fig 5). Tumour/hyaline droplets were also seen at many areas (Table/Fig 4)c,d,(Table/Fig 5)a. Ayoub-Shklar and periodic acid Schiff staining did not reveal ghost cells and mucous cells respectively. A final diagnosis extrafollicular cystic AOT was rendered. OPG taken on the seventh month after enucleation showed adequate healing and regeneration of lingual plate (Table/Fig 6). The patient is under regular follow-up for past 18 months.

Discussion

Odontogenic tumours are heterogenous pathologies derived from dental apparatus (1),(2). AOT, also known as ‘two-third tumour’ is an uncommon epithelial odontogenic tumour accounting for less than 5% of all odontogenic tumours, most commonly seen in association with impacted maxillary canines; the follicular type (3). This variant radiographically resembles DC, and in fact, around 77% of follicular AOTs are diagnosed as DC initially (4). In one of the earliest literature reference, Harbitz F reported “cystic Adamantoma” in the year 1915 which was most probably the cystic presentation of AOT (4). Extra-follicular subtypes are comparatively rare, seen unassociated with impacted teeth, and may be seen above, in between, superimposed to or unrelated to the roots of the teeth. The nature of AOT has been a never-ending debate. While some authorities consider it as a true neoplasm of odontogenic origin, others consider it as hamartoma with their own solid reasons (5). Regardless, as of now, it is considered as a benign neoplasm and is classified under benign epithelial category, even in the latest World Health Organisation (WHO) classification of head and neck tumours (6).

Cystic degeneration in solid tumours is not an uncommon histological feature and is commonly seen in many epithelial and mesenchymal tumours such as Pindborg tumour, cemento-ossifying fibroma and schwannoma to name a few (6),(7),(8),(9),(10). Extensive degeneration is not unusual in ameloblastoma also. Literature search revealed 30 reported cases of cystic AOT; DC is the most commonly associated cyst (11),(12),(13). The aim of the present paper is to report a unique case of cystic variant of AOT with special emphasis on the current understanding.

Gadewar DR and Srikant N described AOT as ‘an intra-cystic proliferation of polygonal and spindle cells’. The term ‘Adenomatoid Odontogenic Cyst (AOC)’ was put forward by Marx and Stern (12). These authors opined that lesion was cystic and the solid nature of AOT was due to intraluminal proliferation of the tumour cells which filled up the cystic space. Further, there are reported cases of AOT that arise either from the lining of DC or Calcifying Odontogenic Cyst (COC); unspecified cystic lining in many cases.

Cystic component is an indispensable histological feature of AOT (Table/Fig 7),(Table/Fig 8); presumably can happen due to pooling of mucoid stroma resulting from the rupture of thin lattice like pattern (mainly seen at the periphery of the tumour close to the capsule) or development of AOT within or adjacent to a pre-existing odontogenic cyst, most commonly DC (5). In the present case, we support the former possibility. As in multiple serial sections, DC like epithelial lining was not noted, neither the lesion was associated with any impacted teeth. The lesion was seen extra-follicularly apical to root apices of mandibular teeth ruling out the origin from pre-existing DC. COC, Glandular Odontogenic Cyst (GOC), Odontogenic Keratocyst (OKC) and unicystic ameloblastoma were also excluded histopathologically.In available literature, description of cystic AOT pertains mainly to AOT seen in association with DC (12),(13). DC develops by accumulation of fluid between the Reduced Enamel Epithelium (REE) and the unerupted tooth crown, and separation of DC from hyperplastic follicle is many a time very subjective (14). The REE is attached to the Cemento-Enamel Junction (CEJ) in contrast to the AOT where the attachment is noted on the radicular portion raising another query whether all the previous reported cases with DC like lining were transformation of DC to AOT or just represents the basaloid cells that form the plexiform pattern. We support the latter hypothesis as in multiple serial sections we also demonstrated bud-like or strand-like extensions into the adjacent stroma.

In contrast to classical AOT, cystic variants were found to be statistically significant larger in size (13), which is explained in the subsequent section. Generally, AOT has an indolent course with no sign of recurrence or persistent growth even after incomplete surgical removal. In one of the earlier analytical reports, Philipsen HP et al., reported a protein content of 5.2 g/100 mL and 7 g/100 mL in one case each of follicular type and extra-follicular type (of AOT) respectively (15). These protein content values were within the range provided for the cystic protein content of non-keratinising odontogenic cysts (5-11 g/100 mL) in contrast to lower range in keratinising cysts (<3.5 g/100 mL) (15). The protein content in the present case was close to the extrafollicular AOT reported previously (16). The values have further been correlated with the nature and direction of expansion (owing to the osmotic pressure), serum osmolarity, lymphatic drainage, nature of globulins and the internal hydrostatic pressure of the cysts. In OKCs, the bony destruction happen in anteroposterior direction at the expense of marrow space while other cysts cause buccal and lingual cortical expansion akin to the present case. This has been widely described in the literature. Definitely, the role of cyst wall cannot be overlooked which through various collagenolytic enzymes, prostaglandins, interleukins, and many more, dictates the clinical behaviour of any lesion. It was demonstrated that in contrary to collagen in the connective tissue wall of radicular cyst and DC, the stroma of AOT consists of poorly packed or pathologic collagen which resembled the connective tissue of OKC wall (17). The extensive expansion and root resorption in the present case, unlike conventional solid AOT with minimal cystic component could thus be due to the cystic degeneration of the solid component with further expansion owing to Toller’s theory and unique connective tissue components of the cyst wall (16).

It is therefore more logical to consider the cystic degeneration as secondary phenomenon. Such lesions may behave aggressively as compared to solid variants and may be regarded as cystic variant of AOT rather than AOC. Furthermore, these lesions should be delineated from the AOT arising in the wall of odontogenic cysts. Cystic variants of AOT are documented in reported cases of follicular and extraosseous AOT. The extra-follicular nature of the lesion in the present case thus supports the existence of cystic variant of AOT.

Conclusion

Although rare, cystic variant of AOT should be considered in the differential diagnosis of radiolucencies of the anterior mandible. Further, this variant deserves delineation from AOT arising from the wall of DCs. The authors believe that cystic degeneration is a secondary phenomenon. Conservative enucleation is the treatment of choice.

References

1.
Pandiar D, Anand R, Kamboj M, Narwal A, Shameena PM, Devi A. Metastasizing ameloblastoma: A 10 year clinicopathological review with an insight into pathogenesis. Head Neck Pathol. 2021;15(3):967-74. [crossref][PubMed]
2.
Pandiar D, Shameena PM, Sudha S, Varma S, Manjusha P, Banyal VS, et al. Odontogenic tumours: A 13year retrospective study of 395 cases in a south Indian teaching institute of Kerala. Oral Maxillofac Pathol J. 2015;6(2):602-08.
3.
Chakraborty R, Sen S, Goyal K, Pandya D. “Two third tumour”: A case report and its differential diagnosis. J Family Med Prim Care. 2019;8(6):2140-43. [crossref][PubMed]
4.
Philipsen HP, Reichart PA, Siar CH, Ng KH, Lau SH, Zhang X, et al. An updated clinical and epidemiological profile of the adenomatoid odontogenic tumour: A collaborative retrospective study. J Oral Pathol Med. 2007;36(7):383-93. [crossref][PubMed]
5.
Rick GM. Adenomatoid odontogenic tumour. Oral Maxillofac Surg Clin North Am. 2004;16(3):333-54. [crossref][PubMed]
6.
Vered M, Wright JM. Update from the 5th Edition of the World Health Organization Classification of Head and Neck Tumours: Odontogenic and Maxillofacial Bone Tumours. Head Neck Pathol. 2022;16(1):63-75. [crossref][PubMed]
7.
Gopalakrishnan R, Simonton S, Rohrer MD, Koutlas IG. Cystic variant of calcifying epithelial odontogenic tumour. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2006;102(6):773-77. [crossref][PubMed]
8.
Barreras CMU, Rivera DQ, Koutlas IG, Cepeda LAG. Clear cell cystic variant of calcifying epithelial odontogenic tumour. Head Neck Pathol. 2014;8(2):229-33. [crossref][PubMed]
9.
Samman AM, Bardeesi AM, Alzahrani MT. Thoracic cystic schwannoma: Case report and review of literature. Spinal Cord Ser Cases. 2021;7(1):7. [crossref][PubMed]
10.
Bouhoute M, Taleb B. Cystic degeneration in cemento-ossifying fibroma: Diagnosis challenge and conservative management- Case report. Int J Surg Case Rep. 2022;90:106676. [crossref][PubMed]
11.
Grover S, Rahim AM, Parakkat NK, Kapoor S, Mittal K, Sharma B, et al. Cystic adenomatoid odontogenic tumour. Case Rep Dent. 2015;2015:503059. [crossref][PubMed]
12.
Gadewar DR, Srikant N. Adenomatoid odontogenic tumour: Tumour or a cyst, a histopathological support for the controversy. Int J Pediatr Otorhinolaryngol. 2010;74(4):333-37.[crossref][PubMed]
13.
Jayasooriya PR, Rambukewella IK, Tilakaratne WM, Mendis BRRN, Lombardi T. Clinico-pathological presentations of cystic and classic adenomatoid odontogenic tumours. Diagnostics (Basel). 2019;10(1):03. [crossref][PubMed]
14.
Paul R, Paul G, Prasad RK, Singh S, Agarwal N, Sinha A. Appearance can be deceptive: Dentigerous cyst crossing the midline. Natl J Maxillofac Surg. 2013;4(1):100-03. [crossref][PubMed]
15.
Philipsen HP, Samman N, Ormiston IW, Wu PC, Reichart PA. Variants of the adenomatoid odontogenic tumour with a note on tumour origin. J Oral Pathol Med. 1992;21(8):348-52. [crossref][PubMed]
16.
Toller PA. Protein substances in odontogenic cyst fluids. Br Dent J. 1970;128(7):317-22. [crossref][PubMed]
17.
Mahajan AM, Mahajan MC, Ganvir SM, Hazarey VK. The role of stroma in the expansion of odontogenic cysts and adenomatoid odontogenic tumour: A polarized microscopy study. J Nat Sci Biol Med. 2013;4(2):316-20.[crossref][PubMed]

DOI and Others

DOI: 10.7860/JCDR/2024/68667.19119

Date of Submission: Nov 19, 2023
Date of Peer Review: Jan 10, 2024
Date of Acceptance: Jan 18, 2024
Date of Publishing: Mar 01, 2024

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: Nov 22, 2023
• Manual Googling: Jan 11, 2024
• iThenticate Software: Jan 17, 2024 (9%)

ETYMOLOGY: Author Origin

EMENDATIONS: 6

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