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Dr Mohan Z Mani

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On Sep 2018




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Prof. Somashekhar Nimbalkar
Head, Department of Pediatrics, Pramukhswami Medical College, Karamsad
Chairman, Research Group, Charutar Arogya Mandal, Karamsad
National Joint Coordinator - Advanced IAP NNF NRP Program
Ex-Member, Governing Body, National Neonatology Forum, New Delhi
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Department of Pediatrics, Pramukhswami Medical College, Karamsad, Anand, Gujarat.
On Sep 2018




Dr. Kalyani R

"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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Sri Devaraj Urs Academy of Higher Education and Research , Kolar, Karnataka
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Dr. Saumya Navit

"As a peer-reviewed journal, the Journal of Clinical and Diagnostic Research provides an opportunity to researchers, scientists and budding professionals to explore the developments in the field of medicine and dentistry and their varied specialities, thus extending our view on biological diversities of living species in relation to medicine.
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Professor and Head
Department of Pediatric Dentistry
Saraswati Dental College
Lucknow
On Sep 2018




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Dr. Arunava Biswas
MD, DM (Clinical Pharmacology)
Assistant Professor
Department of Pharmacology
Calcutta National Medical College & Hospital , Kolkata




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Best regards,
C.S. Ramesh Babu,
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Muzaffarnagar Medical College,
Muzaffarnagar.
On Aug 2018




Dr. Arundhathi. S
"Journal of Clinical and Diagnostic Research (JCDR) is a reputed peer reviewed journal and is constantly involved in publishing high quality research articles related to medicine. Its been a great pleasure to be associated with this esteemed journal as a reviewer and as an author for a couple of years. The editorial board consists of many dedicated and reputed experts as its members and they are doing an appreciable work in guiding budding researchers. JCDR is doing a commendable job in scientific research by promoting excellent quality research & review articles and case reports & series. The reviewers provide appropriate suggestions that improve the quality of articles. I strongly recommend my fraternity to encourage JCDR by contributing their valuable research work in this widely accepted, user friendly journal. I hope my collaboration with JCDR will continue for a long time".



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.
The journal has a monthly publication and the articles are published quite fast. In time compared to other journals. The on-line first publication is also a great advantage and facility to review one's own articles before going to print. The response to any query and permission if required, is quite fast; this is quite commendable. I have a very good experience about seeking quick permission for quoting a photograph (Fig.) from a JCDR article for my chapter authored in an E book. I never thought it would be so easy. No hassles.
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.
My best wishes to Dr. Hemant Jain and all the editorial staff of JCDR for their untiring efforts to bring out this journal. I strongly recommend medical fraternity to publish their valuable research work in this esteemed journal, JCDR".



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.
Writing is the representation of language in a textual medium i e; into the words and sentences on paper. Quality medical manuscript writing in particular, demands not only a high-quality research, but also requires accurate and concise communication of findings and conclusions, with adherence to particular journal guidelines. In medical field whether working in teaching, private, or in corporate institution, everyone wants to excel in his / her own field and get recognised by making manuscripts publication.


Authors are the souls of any journal, and deserve much respect. To publish a journal manuscripts are needed from authors. Authors have a great responsibility for producing facts of their work in terms of number and results truthfully and an individual honesty is expected from authors in this regards. Both ways its true "No authors-No manuscripts-No journals" and "No journals–No manuscripts–No authors". Reviewing a manuscript is also a very responsible and important task of any peer-reviewed journal and to be taken seriously. It needs knowledge on the subject, sincerity, honesty and determination. Although the process of reviewing a manuscript is a time consuming task butit is expected to give one's best remarks within the time frame of the journal.
Salient features of the JCDR: It is a biomedical, multidisciplinary (including all medical and dental specialities), e-journal, with wide scope and extensive author support. At the same time, a free text of manuscript is available in HTML and PDF format. There is fast growing authorship and readership with JCDR as this can be judged by the number of articles published in it i e; in Feb 2007 of its first issue, it contained 5 articles only, and now in its recent volume published in April 2011, it contained 67 manuscripts. This e-journal is fulfilling the commitments and objectives sincerely, (as stated by Editor-in-chief in his preface to first edition) i e; to encourage physicians through the internet, especially from the developing countries who witness a spectrum of disease and acquire a wealth of knowledge to publish their experiences to benefit the medical community in patients care. I also feel that many of us have work of substance, newer ideas, adequate clinical materials but poor in medical writing and hesitation to submit the work and need help. JCDR provides authors help in this regards.
Timely publication of journal: Publication of manuscripts and bringing out the issue in time is one of the positive aspects of JCDR and is possible with strong support team in terms of peer reviewers, proof reading, language check, computer operators, etc. This is one of the great reasons for authors to submit their work with JCDR. Another best part of JCDR is "Online first Publications" facilities available for the authors. This facility not only provides the prompt publications of the manuscripts but at the same time also early availability of the manuscripts for the readers.
Indexation and online availability: Indexation transforms the journal in some sense from its local ownership to the worldwide professional community and to the public.JCDR is indexed with Embase & EMbiology, Google Scholar, Index Copernicus, Chemical Abstracts Service, Journal seek Database, Indian Science Abstracts, to name few of them. Manuscriptspublished in JCDR are available on major search engines ie; google, yahoo, msn.
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.
It is well said that "happy beginning is half done" and it fits perfectly with JCDR. It has grown considerably and I feel it has already grown up from its infancy to adolescence, achieving the status of standard online e-journal form Indian continent since its inception in Feb 2007. This had been made possible due to the efforts and the hard work put in it. The way the JCDR is improving with every new volume, with good quality original manuscripts, makes it a quality journal for readers. I must thank and congratulate Dr Hemant Jain, Editor-in-Chief JCDR and his team for their sincere efforts, dedication, and determination for making JCDR a fast growing journal.
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 : 2023 | Month : January | Volume : 17 | Issue : 1 | Page : ZD08 - ZD10 Full Version

Lymphangiectasia Mimicking Lichenoid Contact Reaction


Published: January 1, 2023 | DOI: https://doi.org/10.7860/JCDR/2023/59312.17255
Vaishnavi Muthukumaran, Aravind Warrier Sankara, CV Divyambika, H Thamizhchelva, N Malathi

1. Postgraduate Student, Department of Oral Medicine and Maxillofacial Radiology, Sri Ramachandra Institute of Higher Education and Research, Chennai, Tamil Nadu, India. 2. Professor and Head, Department of Oral Medicine and Maxillofacial Radiology, Sri Ramachandra Institute of Higher Education and Research, Chennai, Tamil Nadu, India. 3. Professor, Department of Oral Medicine and Maxillofacial Radiology, Sri Ramachandra Institute of Higher Education and Research, Chennai, Tamil Nadu, India. 4. Professor and Head, Department of Oral Pathology and Microbiology, Sri Ramachandra Institute of Higher Education and Research, Chennai, Tamil Nadu, India. 5. Professor, Department of Oral Pathology and Microbiology, Sri Ramachandra Institute of Higher Education and Research, Chennai, Tamil Nadu, India.

Correspondence Address :
Dr. Aravind Warrier Sankara,
1, Mount Poonamallee Road, Sri Ramachandra Nagar, Chennai, Tamil Nadu, India.
E-mail: dentalwarrier@gmail.com

Abstract

Lymphangiectasia, otherwise known as acquired lymphangioma, is unusual to occur in the oral cavity. Among the frequently recognised forms of congenital and acquired entities, the acquired form of the lymphangiectasia may be caused due to trauma or obstruction to the lymphatic system. Commonly, this condition is asymptomatic; however, the needs of aesthetics and functions warrant treatment. Hereby, authors present a case report of a 55-year-old male patient, who came with a chief complaint of burning sensation in his left cheek region for the past three weeks. Clinically, silver amalgam restoration was evident in relation to the tooth numbers 26,27,36 and 37. Soft tissue examination revealed evidence of greyish radiating striae interspersed with hyperpigmentation on the left buccal sulcus and left lateral border of the tongue. Considering the clinical features, a working diagnosis of lichenoid contact reaction was proposed. Further, an incisional biopsy was performed. Histopathological examination showed thin stratified squamous epithelium with vacuolar spaces, which strongly supported the diagnosis of lymphatic malformation. The patient was symptomatically managed with topical triamcinolone acetonide 0.1% twice daily for two weeks. At two weeks follow-up, the patient was symptom-free and hence, the medication was withdrawn. The patient is currently under regular follow-ups.

Keywords

Burning sensation, Greyish radiating striae, Hyperpigmentation, Silver amalgam restoration

Case Report

A 55-year-old male reported to the Department of Oral Medicine and Radiology with a chief complaint of burning sensation in the left inner cheek region for the past three weeks. Initially, burning sensation was only after intake of spicy food which gradually increased with time. Medical history was non contributory. On general examination, all vitals were normal. On local examination, intraorally, greyish radiating striae interspersed with hyperpigmentation was evident on the left buccal sulcus alongside 36, 37, 38 which was non scrapable and lesion was non tender (Table/Fig 1)a. Similar diffuse hyperpigmentation was evident on the left lateral border of ventral surface of the tongue sparing the tip (Table/Fig 1)b. Using Visual Analogue Scale (VAS) the burning pain score was denoted as 5 (1). Contributing the soft tissue finding, silver amalgam restoration was evident in relation to the tooth numbers 26, 27, 36 and 37. On lymph node examination, no abnormality was detected.

Based on this clinical presentation, a provisional diagnosis of lichenoid contact reaction, either due to amalgam restoration or betel quid, was considered. Tobacco-induced hyperpigmentation, postinflammatory pigmentation and oral melanotic macule were considered as other differential diagnosis.

After obtaining the informed consent, routine haematological investigations were performed, which were within normal range and incisional biopsy was done in the left buccal mucosa. Histopathological examination revealed thin stratified squamous epithelium with vacuolar spaces and juxtaepithelium showed plenty of dilated endothelial lined lymphatic channels, deeper section showed the presence of dense fibrosis with few inflammatory cells (Table/Fig 2)a,b. Correlating the clinical and histopathological observation, a final diagnosis of lymphangiectasia involving the left lateral border of tongue and left buccal mucosa was arrived at.

The patient was symptomatically treated with topical triamcinolone acetonide, 0.1% twice daily for two weeks. Meanwhile, all his silver amalgam restorations were replaced with Glass Ionomer Cement (GIC). He was also advised to stop the habit of betel quid pouching and counselled for tobacco cessation. At two weeks, follow-up the patient was symptom-free with VAS score 0 (Table/Fig 3)a,b (1). Medication was stopped and the patient was reassured and explained about the complications associated with this lesion The patient was asymptomatic at six months of follow-up (Table/Fig 4)a,b and is under regular monthly review.

Discussion

Lymphangiectasia also called as acquired lymphangioma develops as a result of an abnormality to previously normal lymphatic drainage due to surgery, trauma, malignancy or radiotherapy (2). The prevalence rate is 4% of all vascular tumours and has neither racial nor gender predilection (3). Lymphangiomas are more common at birth or developed within 20 years of age, due to improper connection of lymphatic channels to the main lymphatic drainage duct, while the acquired form often presents in adulthood as a sequelae of chronic lymphoedema, that leads to disruption as these cells do not anastomose efficiently with bigger lymphatic vessels, provoking areas of lymphatic blockage (4),(5). Approximately, half of all lesions are detected at birth and 90% develop by the age of two years (6).

The site of occurrence is often the head and neck region. Considering the oral cavity, most common site includes anterior two-thirds of the tongue, but infrequent occurrence has been reported in the buccal mucosa, lips, gingiva, palate and alveolar ridge of the mandible (4),(6). Superficial lesion represents papillary lesions with pebbly surface due to the occurrence of several translucent vesicles with same colour as that of adjacent mucosa or occasionally with a mild reddish hue. Interestingly, these give a tapioca pudding or frog eggs-like appearance (4),(7). The present case also presented in buccal mucosa and floor of mouth as a pigmented macule with a reticular pattern, unlike the usual exophytic growth, which has been reported in the literature to be relatively rare oral presentation (8),(9). The unilateral presentation of greyish radiating striae interspersed with hyperpigmentation mimicking lichenoid contact reaction was solely noted, which is the unique feature in this case; and to the best of authors’ knowledge, first to be reported in the literature.

Mucosal pigmentation can be caused by long-term inflammatory mucosal disorders such as oral lichen planus, pemphigus, or pemphigoid (10). The pathophysiology of postinflammatory pigmentation is unknown; however, it is more common in dark-skinned people (11). Multiple brown-black pigmented patches are observed clinically proximal to reticular, erosive or vesicular lesions. Microscopically, melanin laden macrophages accumulate in the superficial connective tissue and there is excessive production of melanin by the melanocytes. Though, the clinical manifestations were consistent with the current instance, the histological findings did not support the diagnosis (10).

The oral melanotic macule is a tiny, well-circumscribed brown-to-black spot that most usually appears on the vermilion zone of the lower lip (33%), followed by the buccal mucosa, gingiva, and palate (6). Histologically, it is marked by enhanced melanin synthesis with normal morphological characteristics. In the current case, both clinical and histological features varied, disqualifying the diagnosis.

In tobacco induced hyperpigmentation, a well-defined and predictable lesion develops at the place where the smokeless tobacco is retained in the oral cavity (12). This type of pigmentation is present in 15% of tobacco chewers and caused by the irritational components in the tobacco, either because of stimulation of melanin generation or due to binding of melanin to the noxious components in the tobacco (12). A similar clinical presentation was noted in the present patient as he had the habit of pouching betel quid in left buccal sulcus, but the absence of microscopic finding of increase melanin synthesis in the basal cell layer ruled out the diagnosis (12). Mixed cell subepithelial infiltration, a deeper diffuse distribution in lamina propria, localised parakeratosis, focal disruption of the granular layer, and cytoid structures in the cornified and granular layers are histological markers of lichenoid lesions. This eliminated the preliminary diagnosis in the current case (13).

The pathogenesis of vessel proliferation in acquired form of lymphatic malformation occurs gradually in stages. Initially, the accumulation of interstitial fluid leads to swelling of the extracellular matrix, to which the endothelial cells lining the lymphatic capillaries become attached; following which the endothelial cells lining the lymph ducts become elongated at the capillary level, β-1-integrin, resulting in activation and phosphorylation of Vascular Endothelial Growth Factor (VEGF) receptor-3 (14),(15). This process results in the proliferation of endothelial cells in the lymph ducts, which is increased by oedema and mechanical pressure (14),(15). It is thought that, the inciting events in the index patient were the oedema caused by repeated trauma to the buccal mucosa and the mechanical pressure exerted by pouching the betel nut. A similar causative factor of trauma associated with trauma due to the mandibular space maintainer was reported previously in a case of lymphangiectasia, mimicking mucocele in lower labial mucosa (16).

Lymphatic vessels with significant dilatations are histopathologic characteristics of lymphangiectasia. The endothelial lining is thin, and the spaces are filled with proteinaceous fluid and lymphocytes. Secondary haemorrhage in lymphatic vessels is likely to occur. Lymphatic fluid, red blood cells, lymphocytes, macrophages and neutrophils are mostly found in the lymphatic spaces. Similar presentation of thin stratified squamous epithelium with vacuolar spaces and plenty of dilated endothelial lined lymphatic channels was noted, affirming the diagnosis (17).

The consequences of deep-seated lesions include obstruction of upper airway, extrusion of tongue, increased salivation, jaw deformity, pain, poor oral hygiene, difficulty in chewing and speaking (14). The management depends upon their type, size, involvement of the structures and infiltration to the surrounding tissues (14). Commonly, employed treatment modalities include laser therapy, cryotherapy, electrocautery, sclerotherapy, radiofrequency ablation and surgical excision (4). Traditionally, treatment entails surgically removal without endangering crucial structures (18). However, some adverse effects such as nerve injuries, recurrence, secondary infections, scar formation and incomplete resections because of involvement of adjacent vital structures (19).

Radiofrequency ablation used in a patient with oral lymphangioma showed no recurrence at one year follow-up. The present authors chose radiofrequency ablation because of the advantages of the technique to reach precise areas of tissue involvement, and being minimally invasive (8). Surgical excision was done for an exophytic growth in a 30-year-old patient, which showed no recurrence at one year follow-up (9). One of the two previously reported oral lymphangiectasia case associated with Crohn’s disease, was treated successfully with short burst cryotherapy (20). Radiation, laser and sclerotherapy have been associated with low success rates with potential side-effects (8). As the index patient had only superficial lesion, the patient was symptomatically managed and explained the probability of adverse outcomes requiring close follow-ups.

Conclusion

Acquired lymphangiectasia is rarely encountered in the oral cavity. Their timely recognition allows commencement of treatment with adequate follow-ups to prevent morbid states and recurrence. The case was exceptional due to the aetiology of trauma and the clinical appearance mirroring the lichenoid contact reaction. Consequently, while addressing such superficial mucosal lesions, acquired lymphangiectasia must be taken into account in the differential diagnosis. Fundamental knowledge about this condition is utmost importance, for its right diagnosis and proper therapeutic indication.

References

1.
Haefeli M, Elfering A. Pain assessment. Eur Spine J. 2006;15 Suppl 1(Suppl 1):S17- 24. Doi: 10.1007/s00586-005-1044-x. Epub 2005 Dec 1. PMID: 16320034; PMCID: PMC3454549. [crossref] [PubMed]
2.
Wege J, Anabtawi M, Blackwell MA, Patterson A. Lymphangioma formation following hyaluronic acid injection for lip augmentation. Cureus. 2021;13(1):e12929. Doi: 10.7759/cureus.12929. PMID: 33654610; PMCID: PMC7910224. [crossref] [PubMed]
3.
Miceli A, Stewart KM. Lymphangioma. 2021 Aug 12. In: StatPearls [Internet]. Treasure Island (FL): StatPearls Publishing; 2022 Jan. PMID: 29261940.
4.
Tayefeh A, Asadollahi A, Saatloo MV. A rare case report of oral lymphangioma in buccal mucosa. Ann Clin Case Stud. 2020;2(4):1035.
5.
Abe A, Kurita K, Ito Y. Acquired lymphatic malformations of the buccal mucosa: A case report. Clin Case Rep. 2018;6(10):1929-32. [crossref] [PubMed]
6.
Neville BW, Damm DD, Allen CM, Bouquot JE (Eds). Oraland maxillofacial pathology (3rd ed). Toronto (ON): WB Saunders Company 2009.
7.
Vasconcelos MG, Santos BC, Lemos LCP, Ribeiro BF, Iglesias DPP, Vasconcelos RG et al. Oral lymphangioma: Case report. RSBO. 2011;8:352-56.
8.
Bozkaya S, Ug? ar D, Karaca I, Ceylan A, Uslu S, Baris¸ E, et al. The treatment of lymphangioma in the buccal mucosa by radiofrequency ablation: A case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2006;102(5):e28-31. [crossref] [PubMed]
9.
Lacin N, Yalcin M, Efeoglu BF, Izol BS, Tünel A. The treatment of lymphangioma in the buccal mucosa by surgical excision: A rare case report. Res Rep Oral Maxillofac Surg. 2018;2:013. [crossref] [PubMed]
10.
Gondak RO, da Silva-Jorge R, Jorge J, Lopes MA, Vargas PA. Oral pigmented lesions: Clinicopathologic features and review of the literature. Med Oral Patol Oral Cir Bucal. 2012;1:17(6):e919-24. [crossref] [PubMed]
11.
Halder RM, Nootheti PK. Ethnic skin disorders overview. J Am Acad Dermatol. 2003;48:S143-48. [crossref] [PubMed]
12.
Taybos G. Oral changes associated with tobacco use. Am J Med Sci. 2003;326(4):179-82. [crossref] [PubMed]
13.
McParland H, Warnakulasuriya S. Oral lichenoid contact lesions to mercury and dental amalgam-a review. J Biomed Biotechnol. 2012;2012:589569. [crossref] [PubMed]
14.
Usha V, Sivasankari T, Jeelani S, Asokan GS, Parthiban J. Lymphangioma of the tongue- a case report and review of literature. J Clin Diagn Res. 2014;8(9):ZD12-14.
15.
Planas-Paz L, Lammert E. Mechanical forces in lymphatic vascular development and disease. Cell Mol Life Sci. 2013;70(22):4341-54. [crossref] [PubMed]
16.
Flores FDA, Rumayor PA, Vértiz FK, Galindo RM, Esparza DSC, Iñigo BA. Acquired lymphangiectasia of lower lip. A clinical case report. Rev Odont Mex. 2020;24(1):66-71.
17.
Sunil S, Gopakumar D, Sreenivasan BS. Oral lymphangioma- Case reports and review of literature. Contemp Clin Dent. 2012;3(1):116-18. [crossref] [PubMed]
18.
Pammar C, Kotrashetti VS, Nayak R, Hosmani J. Lymphangioma of the buccal mucosa: A case report with a literature review. J Orofac Sci. 2015;7:129-31. [crossref]
19.
Mandel L. Parotid area lymphangioma in an adult: Case report. J Oral Maxillofac Surg. 2004;62(10):1320-23. [crossref] [PubMed]
20.
Galvin S, Flint SR, Toner ME, Healy CM, Ekanayake K. Oral lymphangiectasias and Crohn’s disease: Two case reports. Oral Surg Oral Med Oral Pathol Oral Radiol. 2018;126(1):e31-e34. [crossref] [PubMed]

DOI and Others

DOI: 10.7860/JCDR/2023/59312.17255

Date of Submission: Jul 26, 2022
Date of Peer Review: Sep 22, 2022
Date of Acceptance: Oct 27, 2022
Date of Publishing: Jan 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 29, 2022
• Manual Googling: Oct 20, 2022
• iThenticate Software: Oct 26, 2022 (16%)

ETYMOLOGY: Author Origin

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