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Dr Mohan Z Mani

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On Sep 2018




Prof. Somashekhar Nimbalkar

"Over the last few years, we have published our research regularly in Journal of Clinical and Diagnostic Research. Having published in more than 20 high impact journals over the last five years including several high impact ones and reviewing articles for even more journals across my fields of interest, we value our published work in JCDR for their high standards in publishing scientific articles. The ease of submission, the rapid reviews in under a month, the high quality of their reviewers and keen attention to the final process of proofs and publication, ensure that there are no mistakes in the final article. We have been asked clarifications on several occasions and have been happy to provide them and it exemplifies the commitment to quality of the team at JCDR."



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
Ex-President - National Neonatology Forum Gujarat State Chapter
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 Medical College
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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Dr Saumya Navit
Professor and Head
Department of Pediatric Dentistry
Saraswati Dental College
Lucknow
On Sep 2018




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"My sincere attachment with JCDR as an author as well as reviewer is a learning experience . Their systematic approach in publication of article in various categories is really praiseworthy.
Their prompt and timely response to review's query and the manner in which they have set the reviewing process helps in extracting the best possible scientific writings for publication.
It's a honour and pride to be a part of the JCDR team. My very best wishes to JCDR and hope it will sparkle up above the sky as a high indexed journal in near future."



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,
Associate Professor of Anatomy,
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 Series
Year : 2022 | Month : June | Volume : 16 | Issue : 6 | Page : ER01 - ER04 Full Version

Uncommon Pathological Presentations of Mature Cystic Teratoma: A Case Series


Published: June 1, 2022 | DOI: https://doi.org/10.7860/JCDR/2022/56624.16462
Suchita Pant, Usha Joshi, Kriti Joshi

1. Assistant Professor, Department of Pathology, Government Medical College, Haldwani, Uttarakhand, India. 2. Associate Professor, Department of Pathology, Government Medical College, Haldwani, Uttarakhand, India. 3. Senior Resident, Department of Pathology, Sawai Man Singh Medical College, Jaipur, Rajasthan, India.

Correspondence Address :
Dr. Suchita Pant,
3-47/2, G.B Pant Marg, Civil Lines, Bhoti Aparao, Haldwani, Uttarakhand, India.
E-mail : suchita.pant786.sp@gmail.com

Abstract

Mature cystic teratomas are benign tumours of the ovary but rarely certain unusual gross and microscopic findings might complicate their diagnosis or may have an impact on their clinical outcome. Here, we are presenting a case series of five unusual pathological presentations of mature cystic teratomas of ovary. Out of five cases, three cases were of collision tumours, in which combination of mature cystic teratoma is seen with mucinous cystadenoma in two cases, and with serous cystadenoma in one case. True collision tumours are defined as histologically distinct neoplasms in the same tissue or organ without any histologic admixture or intermediate cell population zone between two components. These tumours are located in various organs but ovarian location is rare. Mature cystic teratoma is the most common component of collision combinations in the ovary. All classes of ovarian tumours, benign, borderline and malignant may collide and therefore, clinical outcomes in collision tumours depend on individual tumour characteristics. The fourth case was of a mature cystic teratoma associated with a distinct haemangiomatous component. Mature cystic teratoma associated with prominent haemangiomatous component is a very rare finding with only few case reports in the literature and it should be differentiated from true ovarian haemangioma, lymphangioma, and angiosarcoma. The fifth case was of bilateral mature cystic teratomas associated with a well-defined lipomatous lesion in right sided mature cystic teratoma mimicking lipoma. Lipomatous ovarian lesions are very rare and mature cystic teratoma with prominent adipocytic proliferation is also an exceedingly uncommon finding and it should be differentiated from true ovarian lipoma and other atypical lipomatous proliferations. Most of these tumours remain clinically and radiologically unrecognised, therefore their histological recognition is essential for adequate patient management.

Keywords

Collision, Cystadenoma, Dermoid cyst, Haemangiomatous, Lipomatous

Mature cystic teratomas are the most common ovarian germ cell neoplasms and constitute 10-20% of all ovarian neoplasms (1). These neoplasms can rarely have unusual gross and microscopic presentations which interferes with their clinical and radiological interpretation and histopathological examination becomes crucial for their correct diagnosis. The cases included in this study presented in the Department of Obstetrics and Gynaecology and were reported in the Department of Pathology, Government Medical College, Haldwani, Uttarakhand, India. Collision tumours are rare neoplasms which are reported in various organs but ovarian location is rare. Mature cystic teratoma is the most common component of collision combinations in the ovary. Three cases of collision tumours are included in this series and all these cases had mature cystic teratoma as one of the colliding components. Another case was of a mature cystic teratoma with florid vascular proliferation, which is an exceedingly rare entity with only a few reported cases in the literature. Lipomatous ovarian lesions are very rare and mature cystic teratoma with prominent adipocytic proliferation is also an exceedingly uncommon finding and it should be differentiated from true ovarian lipoma and other atypical lipomatous proliferations. One case of an adipocyte rich teratoma is included in this series to highlight this rare presentation.

Case Report

Case 1

First case was of a 24-year-old female, unmarried, who presented with complaints of abdominal pain since five months. Physical examination revealed right sided adnexal mass. Her medical history was non contributory. The Ultrasonography (USG) findings revealed a cystic multilocular mass originating from the right ovary. Laparoscopic cyst removal was done with a provisional diagnosis of right-sided mucinous cystadenoma. Gross examination showed cystectomy specimen measuring 6.0×5.0×2.0 cm. Wall thickness was 0.1-0.2 cm. External surface was smooth. Cut surface showed multiloculated cyst, filled with mucinous material. Another area in the cyst showed pultaceous material and tufts of hair. Differential diagnosis considered were mature cystic teratoma, collision tumour and composite tumour, based on gross examination. On microscopy, sections from multiloculated cyst revealed fibrocollagenous cyst wall lined by gastric foveolar type of epithelium. Sections from another area depicted keratinised stratified squamous epithelium with underlying pilosebaceous units and adipose tissue. These two components were discrete with no histological admixture, therefore a final diagnosis of collision tumour comprising of mature cystic teratoma and mucinous cystadenoma was made (Table/Fig 1)a,b.

Case 2

Second case was of a 43-year-old female patient, who presented with an abdominal lump and menstrual irregularity for the past 8 months. She was Para 2, Living 2 (P2L2) and her past medical history was unremarkable. Physical examination revealed left side adnexal mass. Magnetic Resonance Imaging (MRI) findings suggested left-sided cystic ovarian mass. Her Cancer Antigen (CA) 125 level was 33 IU/mL. Provisional diagnosis of left sided dermoid cyst was made. Laparotomy was done with left-sided salpingo-oophorectomy. Gross examination showed salpingo-oophorectomy specimen measuring 18.0×15.0×13.0 cm with attached fallopian tube measuring 4.0×0.8 cm. External surface was smooth. On cut, a loculated cyst was identified, filled with the clear serous fluid. A smaller cyst was present within cystic cavity measuring 2 cm in maximum dimension and filled with pultaceous material and hair. Representative sections were taken. On microscopy, sections from the larger cyst revealed fibrocollagenous cyst wall lined by low cuboidal ciliated epithelium. Sections from the smaller cyst showed keratinised stratified squamous epithelium with underlying tissue displaying sebaceous glands, respiratory type epithelium and collections of seromucinous glands. These two components were discrete with no histological admixture. Based on these histological findings, a diagnosis of collision tumour comprising of dermoid cyst and serous cystadenoma was established (Table/Fig 2)a,b.

Case 3

A 41-year-old female, presented with complaints of lower abdominal pain, heaviness and menstrual irregularity since 9 months. She was P1L1 and her past medical history was non contributory. Radiological investigation had suggested the diagnosis of fibroid uterus with a cystic, multilocular, and non vascularised formation in the right ovary. Total Abdominal Hysterectomy (TAH) with bilateral salpingo-oophorectomy was done. Right sided ovarian cyst measured 9.0×7.0×3.0 cm. External surface was lobulated. Cut surface revealed loculated cyst filled with mucinous material. One cystic cavity was filled with pultaceous material and hair. Multiple sections taken from cystic component depicted features of benign mucinous cystadenoma, whereas sections from solid pultaceous component exhibited features of mature cystic teratoma. Thus, establishing the diagnosis of collision tumour, comprising of mucinous cystadenoma and mature cystic teratoma (Table/Fig 3)a,b.

Case 4

Fourth case was of a 37-year-old female patient, who presented with off and on lower abdominal pain since one year. Her past medical history and laboratory investigations were unremarkable. Physical examination revealed a right sided adnexal mass. The USG revealed right sided ovarian semicystic mass measuring 9.6×6.0×3.0 cms.There was no sign of ascites on USG examination. Her CA-125 level was 30 IU/L. Laparotomy was done with right sided ovarian cystectomy. Gross examination showed cystectomy specimen measuring 9.0×5.0×2.5 cm. External surface was smooth. Cut surface was partly solid and partly cystic. On microscopy, sections studied from cystic area revealed cyst wall lined by stratified squamous epithelium with luminal keratinous flakes. Sections from solid area showed a relatively circumscribed lesion depicting florid proliferation of dilated and congested vascular channels, lined by flattened endothelium, embedded in a fibrocollagenous stromal tissue. On extensive sampling from solid area, focal areas lined by stratified squamous epithelium were also identified. On the basis of these findings a diagnosis of mature cystic teratoma with prominent haemangiomatous component was made (Table/Fig 4)a,b.

Case 5

Fifth case was of a 41-year-old female, who presented with abdominal lump, abdominal pain and weakness for the past four months. Her medical history was unremarkable with regular menstrual cycle and no signs of hyperandrogenic state were seen. Physical examination revealed bilateral adnexal mass. The USG revealed solid cystic bilateral adnexal mass. Gross examination showed right sided salpingo-oophorectomy specimen and left sided cystectomy specimen. Right sided ovarian cyst was 5.0×4.0×3.0 cm with attached fallopian tube measuring 4.8×0.7 cm. External surface was smooth. Cut surface showed cystic cavity filled with pultaceous material and hair. At one place a well-defined solid area was identified measuring 2.0×1.8 cm. On cut, it was homogeneous and yellowish in colour. Left cystectomy specimen was 7.4×6.0×4.1 cm. On cut, it was filled with pultaceous material and hair. Residual ovary was identified at one focus. Sections taken from representative areas. On microscopy, sections from bilateral cysts showed features of mature cystic teratoma. Sections taken from the well-defined solid area in right-sided cyst revealed a well circumscribed lesion comprising of lobules of mature adipose tissue, separated by thin fibrous septa and few benign eccrine ducts were seen at the periphery of the lesion. Based on gross and microscopic evaluation a diagnosis of bilateral mature cystic teratoma with a distinct right sided adipocyte rich teratoma was made (Table/Fig 5)a,b.

Discussion

True collision tumours are defined as histologically distinct neoplasms in the same tissue or organ without any histologic admixture or intermediate cell population zone between two components, whereas there is histological intermixing of different components in composite tumours (2). These tumours are located in various organs but ovarian location is rare. The biological behaviour of each component in a collision tumour depends on individual tumour characteristics, therefore their recognition is important for further management.

Various combinations have been reported in collision tumours of the ovary such as mature cystic teratoma and mucinous cystadenoma (3), serous cystadenocarcinoma and teratoma (4), carcinosarcoma and dermoid (5), mature cystic teratoma and fibroma (6).

Many theories have been proposed explaining the origin of collision tumour. According to the first proposed theory, coexistence of two primary tumours in the same tissue or organ is due to a “chance accidental meeting” (7). The second theory states that the first tumour present at the primary site leads to some changes in the microenvironment causing the development of second primary tumour or facilitates the seeding of metastatic tumour cells. The third hypothesis considers the possibility of origin of different components from a common stem cell (8). Few other hypotheses regarding origin of collision tumour include surface metaplasia and teratomatous origin theory. Ultrastructural and mucin histochemical studies support the surface metaplasia theory (9),(10). Whereas, frequent co-existence of mature cystic teratoma with mucinous cystadenoma forms the basis for teratomatous origin theory. As mucinous element in mature cystic teratoma is more commonly of intestinal type rather than Mullerian, this again supports the teratomatous origin of mucinous tumours. Fuji K et al., had also demonstrated similar genetic pattern in both mucinous and teratomatous components, further attesting to the teratomatous origin theory (11).

Three cases in this series are diagnosed as collision tumours; one case had a combination of mature cystic teratoma with serous cystadenoma and two cases had mature cystic teratoma with co-existing mucinous cystadenoma. The cystadenoma component was colliding with mature cystic teratoma in all the three cases, and no admixture of two components or any intermediate cell zone was identified on extensive sampling, thus excluding the possibility of composite tumour and establishing the diagnosis of collision tumour (Table/Fig 6).

Mature cystic teratomas associated with prominent haemangiomatous component is a very rare finding with only few case reports in the literature (12),(13). The pathological differentials for this phenomenon are true ovarian haemangioma, prominent haemangiomatous component of a teratoma, lymphangioma and angiosarcoma (Table/Fig 6). However, in the present case, no nuclear pleomorphism, mitosis or necrosis was seen excluding the diagnosis of angiosarcoma and also there was absence of pale eosinophilic secretions in vascular lumens which ruled out the possibility of lymphangioma. Other entities which should be excluded are proliferating blood vessels in hilar region and closely arranged medullary blood vessels in a post-menopausal female, though these physiological changes in ovary do not form a mass lesion and are usually admixed with nerve fibres and lymphatics (14). Vascular lesions of ovary is a highly debatable topic and some authors had earlier considered pure ovarian haemangioma as monodermal teratoma. However, this concept was challenged by Prus D et al., when they established on the basis of molecular genetic tests that ovarian haemangioma associated with mixed germ cell tumour in their case of Turner syndrome was of somatic origin and not germ cell derived (15). In the present case, cystic teratomatous area was present separately from the greyish brown solid haemangiomatous component raising the possibility of mature cystic teratoma associated with cavernous haemangioma. However, on further sampling of solid component, focal areas lined by stratified squamous epithelium were identified, thus establishing the diagnosis of mature cystic teratoma with a prominent haemangiomatous component. Therefore, in a case of teratomas with prominent vascular proliferations, extensive sampling is required to unearth other teratomatous components, as their presence would rule out the possibility of true ovarian haemangioma.

Adipose tissue is not native to the ovary and lipomatous ovarian lesions are exceedingly rare. Various theories explaining the histogenesis of lipomatous lesions in the ovary are:

• Misplaced embryonic fat cells

• Adipose metaplasia of ovarian mesenchymal cells

• Lipomatous lesions originating from fat cells of teratoma (16)

In the present case, possibilities of ovarian lipoma, adipocyte rich teratoma and atypical lipomatous tumour were considered (Table/Fig 6). However, this lipomatous lesion was associated with adjacent mature cystic teratoma and also few eccrine ducts were present at the periphery of the lesion [Table/Fig-5b], thus excluding the possibility of pure ovarian lipoma. No atypical hyperchromatic nuclei or lipoblasts were seen even on extensive sectioning, which ruled out the possibility of atypical lipomatous tumour, and thus establishing the diagnosis of a separate adipocyte rich teratoma.

Gardella C et al., (17), had reported a case of ovarian lipomatous tumour. However, in their case the presence of benign sweat ducts at the periphery of ovarian lipoma, was indicative of teratomatous origin of lipomatous tumour. Mpatsoulis D et al., had reported prominent adipocytic differentiation in mature cystic teratoma associated with Leydig cell hyperplasia and clinical manifestations of hyperandrogenism. The authors contended that adipose tissue is a complex endocrine organ and might be at least partially responsible for androgenic manifestations (18). However, no evidence of hyperandrogenic state or Leydig cell hyperplasia was associated with lipomatous proliferation in the present case. Therefore, we conclude that this discrete lipomatous lesion in our case is a separate non functioning adipocyte rich teratoma.

The factors which affect management and prognosis of these tumours include types of colliding or associated components, most aggressive component present, and stage of malignant tumour, if malignant component is present (19). However, in all these cases mature cystic teratomas were associated with benign lesions, thus surgical excision of affected ovary was considered curative.

Conclusion

The cases included in this series had very uncommon pathological presentations of mature cystic teratomas, and they were diagnosed incidentally after histopathological evaluation only. Therefore, extensive tissue sampling of cystic ovarian masses is highly recommended to avoid a possible misdiagnosis and to unearth the buried components, as this might impact patient management and outcome.

References

1.
Beker JS, Hacker NF, Hengst TC. Practical gynecologic oncology. Fourth ed. Philadelphia: Lippincott Williams and Wilkins. 2005. pp.443.
2.
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DOI and Others

DOI: 10.7860/JCDR/2022/56624.16462

Date of Submission: Mar 24, 2022
Date of Peer Review: Apr 7, 2022
Date of Acceptance: May 13, 2022
Date of Publishing: Jun 01, 2022

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: Mar 28, 2022
• Manual Googling: May 12, 2022
• iThenticate Software: May 16, 2022 (4%)

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