Journal of Histopathology and Cytopathology

Official Organ of Bangladesh Academy of Pathology

Vol 4, No 2, July 2020

Front Cover PDF

Index/Contents PDF

Inside Back cover PDF

Contents

Information for Contributors

General Information

The Journal of Histopathology and Cytopathology (JHC) aims in our understanding of the pathophysiological and pathogenetic mechanisms of human disease by publishing original papers, review articles, case reports and short communications related to basic and translational fields in pathology. It serves as bridges between basic biomedical science and clinical medicine with particular emphasis on, but is not restricted to, tissue based studies only. It is published twice a year as the Journal Committee of the Bangladesh Academy of Pathology.

Manuscript Preparation.

Manuscripts should be prepared in MS Word format in accordance with The Uniform Requirements for Manuscripts Submitted to Biomedical Journals

(see http://www.icmje.org). All pages of the manuscript should be double-spaced and numbered consecutively beginning with the Title page.  Each of the following sections should begin on separate pages: Title,  Name and affiliation of authors, Abstract and Keywords, Text, Acknowledgements, References, individual Tables and legends.  Reformatting of the accepted papers may be needed according to the Journal specifications.

Title Page

The title page should include (i) type of publication (original, review, case report etc.) (ii) the complete title of the article (iii) authors’ name in abbreviation  (iv) list of authors including full name, highest degree, signature, designation and institutional affiliation and (v) name, mailing address, email and telephone/mobile number of author responsible for correspondence.

Abstracts

It should begin with full title of the article. Do not write authors name in the abstract page. The abstracts should not be more than 200 words. The abstract should state the purpose of the study or investigations, basic procedures, main findings and principal conclusion. Three to ten keywords may be provided below the abstract using terms from the Medical Subject Headings (Index Medicus, NLM, USA). Abbreviations and citations should be avoided.

 Text

The text of the original articles should be divided into following sections: Introduction,

Methods, Result and Discussion.

 References

References to literature should be numbered in Arabic numerical in superscripts

consecutively in the order in which they are mentioned in the text. At the end of article the full list of references should give the name of all authors followed by the title of the article, the title of the journal abbreviated according to Index Medicus, the year of publication, volume number and first and last pages of the article. Title of the books should be followed by the edition, place of publication, the publisher, the year and the relevant pages. Examples of correct form of reference are given below: References should begin on a new page, be double-spaced and numbered in order of citation in the text, including citations in tables and figure legends. Citations that first appear in tables, figures, or supplemental data should be numbered according to the item’s first call out in the text; a separate reference list should not be prepared for supplemental data. Complete author citation is required (use of “et al” is only acceptable for sources with more than 35 authors).

References should conform to the style of the Journal.

 Examples follow:

Journals: van Riel D, Leijten LM, Kochs G, Osterhaus AD, Kuiken T: Decrease of Virus Receptors during Highly Pathogenic H5N1 Virus Infection in Humans and Other Mammals. Am J Pathol 2013, 183:1382-1389

 Electronic Journals: Moher D, Liberati A, Tetzlaff J, Altman DG, The PRISMA Group: Preferred Reporting Items for Systematic Reviews and Meta-Analyses: The PRISMA Statement. PLoS Med 2009, 6:e1000097. http://dx.doi.org/10.1371/journal.pmed.1000097

 Books: Frosch MP: Central Nervous System. Robbins Basic Pathology, 9th Edition. Edited by Kumar V, Abbas AK, Aster JC. Philadelphia, PA, Saunders, 2012, pp. 811-850

 Product Inserts: Cite in text only: (Affymetrix technical note: Globin Reduction Protocol: A Method for Processing Whole Blood RNA Samples for Improved Array Results. Santa Clara, CA).

 Web sites: Cite in text only. See Data Supplements and Non-Traditional Media section below for proper use of web site references. Use the doi when available. Include the name of the institution sponsoring the web site, URL address with direct linkage to the referenced information, and date of last access.

Tables

Tables should be typed written on separate numbered pages submitted after the main text on separate pages, as part of the manuscript. The preferred file format for Tables is MS Word. and should follow the reference list. All tables should be numbered consecutively using Roman numerical. Each must carry a brief descriptive heading. Tables should be planned to fit within print area. Table footnotes should use the sequential symbols: *, †, ‡, §, ¶, ∥; and abbreviations.

Illustrations

Figure file formats (including those embedded in the text) are unacceptable.

Photographs and photomicrographs should be of high resolution (minimum 5 mega pixels), in original unedited form and jpg format. These should contain a legend with magnification and stain used. Figure number and name of the first author should be mentioned in each file. Legend should be given in separate page.  Patients’ identification should be hidden.

Abbreviation

Standard abbreviation should be used whenever possible. The full term for which the abbreviation stands followed by abbreviation in parenthesis should be proceed

the first use of the abbreviation in the text except for standard units of measurements

like 27^OC and 25 mmol/L etc.

Letters to the editor

Communications with reference to an article published in the journal and current health

problems in the community will be accepted as letter to the editor.

 Electronic Copy

An electronic copy (soft copy) in the form of CD must be submitted with the printed copy of the article. Electronic copy may be send by email attachment at sadequel@yahoo.com. Text should be processed with MS Word and pictures should be saved in JPG format.

Manuscript Submission

Electronic version of the manuscripts should be submitted through email to the Executive Editor.  Alternatively send DVD/CD to: The Executive Editor of the Journal of Histopathology and Cytopathology.  A cover letter to the editor must accompany the manuscript stating any,  (a) conflicts of interest (both financial and personal), (b) that the manuscript has not been published previously and is not being considered concurrently by another publication, and (c) all authors and acknowledged contributors have read and approved the manuscript. Submissions are not considered for review if previously published in any form (print or online) other than as an abstract. The editor reserves the customary right to style and if necessary shorten the material accepted for publication and to determine the priority and time of publication. Editor assumes that work based on honest observations. It is not the task of the editor to investigate scientific fraud paper.

Proofs

The corresponding author will be contacted by email once proofs are ready, and will be directed to download electronic proofs from a secure website. The author should check the proofs carefully, mark any printer’s errors, and answer queries as requested. Author changes should be kept to a minimum. Proof corrections and replacement figures (if any) must be returned within 48 hours to avoid any delay in publication.

The Review Strategy

On receipt, manuscripts are assessed by the Editor-in-Chief, to one Associate Editor. The Reviewers’ and Associate Editor’s views are used by the Editor-in-Chief (or a Senior Editor) in reaching a decision, usually within three weeks of submission.

 Summary of Submission, General points

Format the word processing document as double spaced A4 pages with an additional space between paragraphs and margins of at least 2 cm all round. Use a 12-pt standard font such as Times, Helvetica or Arial (with Symbol for special characters). Do not use line numbering, but include page numbers in the header or footer, aligned right. Use consistent, preferably UK English spelling.

 Manuscript title

This should be clear, simple and concise; long titles lack impact. Please remember that many readers will only scan titles, so they should reflect the message of the paper and catch the readers’ attention.

 A short running title

This must be 75 characters or less, including spaces, and reflect the main title and content of the manuscript.

 List of authors

Authorship credit should be based only on 1) substantial contributions to conception and design, or acquisition of data, or analysis and interpretation of data; 2) drafting the article or revising it critically for important intellectual content; 3) final approval of the version to be published.

 A statement outlining the specific contribution of each author to the manuscript and the work reported in it must appear after the acknowledgements section (see below).

 Full affiliations of all authors:

Include the name of the department(s) and institution(s) to which the work should be attributed. Append the corresponding author(s) full postal address, phone number and email address.

 Conflict of interest statements

Authors must disclose all financial and personal relationships that might bias their work; to prevent ambiguity, a conflict of interest statement must appear on the manuscript title page, detailing any conflicts (or the absence thereof) for each author.

 Word count (from beginning of Introduction to end of Discussion)

Concise articles make a greater impact than long ones and are less likely to be delayed by editing to a suitable length. Full articles should be no more than 4000 words from the beginning of the Introduction to the end of the Discussion. Review articles and special features may occasionally exceed this limit by arrangement with the Editor-in-Chief.

 Abstract (not structured and no more than 300 words)

Following the title page(s), the next page should carry an unstructured prose abstract of 300 words or less. It should clearly convey the purposes of the study, and the main procedures, findings and conclusions. It should be understandable without reference to the rest of the paper, and contain no citation to references in the reference list. Only standard abbreviations as listed below are permitted.

 Keywords (3 to 10)

Below the abstract, authors should provide and identify as such 3 to 10 keywords or short phrases to assist indexing the article and that may be published with the abstract. MESH headings are a useful guide for authors in considering keywords.

 Manuscript structure

Research articles are divided into sections with the headings: Abstract, Introduction, Methods, Results and Discussion. Long articles may need subheadings (especially within the Results and Discussion) to clarify their content. The sections should not be numbered. Other types of articles, such as reviews and commentaries, still need a title and abstract and should adhere as closely as possible to these guidelines.

Epidermal Nevus with Epidermolytic Hyperkeratosis: A Case Report

*Fatima K,¹ Banu SG,² Kamal M³

1. *Dr. Kaniz Fatima, Resident, Phase-B, Department of Pathology, Bangabandhu Sheikh Mujib Medical University (BSMMU).joty2005.kfz@gmail.com
2. Sultana Gulshana Banu, Associate Professor, Department of Pathology, Bangabandhu Sheikh Mujib Medical University (BSMMU).
3. Mohammed Kamal, Professor, Department of Pathology, Bangabandhu Sheikh Mujib Medical University (BSMMU).

*Correspondence.

Abstract

Epidermal nevus has various histological patterns. Epidermal nevus having features of epidermolytic hyperkeratosis is a rare condition. This lesion is clinically indistinguishable from other epidermal nevi but shows characteristic histological features. Diagnosis of this entity is important management. Epidermolytic hyperkeratosis is an autosomal dominant disease. Offspring of these patients may have generalized epidermolytic hyperkeratosis. Here we present a case of 12 years old boy with Epidermal nevus with epidermolytic hyperkeratosis, which is a rare entity.

[Journal of Histopathology and Cytopathology, 2020 Jan; 4 (1):65-69]

 Keywords: Epidermal nevus, Epidermolytic hyperkeratosis, Generalized epidermolytic hyperkeratosis, Line of Blaschko.

 Introduction

Epidermal nevus comprises a heterogenous group of diseases. It is a congenital non-inflammatory cutaneous hamartoma. It may occur sporadically or as a part of several syndromes. The condition affects 1 in 1000 people in the world.¹ Histologically, epidermal nevi are composed of keratinocytes, apocrine glands, eccrine glands, sebaceous glands or other components of pilosebaceous unit. Epidermal nevi are traditionally asymptomatic. Small number of cases of epidermal nevi show histologic features of epidermolysis hyperkeratosis. Epidermolytic hyperkeratosis has been observed in variety of benign and malignant skin condition or hereditary disorders. Epidermal nevus with epidermolysis hyperkeratosis has a significant clinical importance. This patient carries the risk of parenting a child of gerneralizedepidermolytichyperkeratosis.² We report a case of epidermal nevus showing epidermolytic hyperkeratosis in a 12 year old boy for the rarity of the entity.

 Case report

A 12 year old boy of a non-consanguineous parentage, presented with non-pruritic, dark coloured elevated skin eruptions since birth. The lesion was first observed over the dorsum of right foot. After that lesions gradually appeared in front of leg and thigh, lower abdomen and flexor aspect of both forearm. Lesions were not related with any seasonal variation. Patient had a normal birth history and developmental milestones. Right sided extremities were more involved than left side.Examination revealed numerous hyperpigmented warty papules distributed in both extremities and lower abdomen (Figure. 1 and 2).  Hair, nails and oral mucosa were normal. Other system examination revealed no abnormality. No laboratory investigation was done.

Clinically it was diagnosised as Linear verrucous epidermal nevus. For histopathological examination 3 mm punch biopsy was taken from right foot. Histological examination revealed hyperkeratosis, acanthosis, papillomatosis, elongated rete ridges. The dermis revealed mild perivascular infiltration of chronic inflammatory cells.Some foci also revealed perinuclear vacuolization of the keratinocytes in spinous and granular layers, and increased number of keratohyalin granules in the stratum granulosum (Figure 3, 4 and 5).  So, histologically it was diagnosed as epidermal nevus with epidermolytic hyperkeratosis.

Discussion

Epidermal nevus is hamartoma of skin, occurs due to over growth of epidermis. It arises from embryonic ectoderm as a result of mosaic postzygotic mutations. Lesions are present at birth in about half of the patients or may develop early in childhood. Depending on the affected component of the epidermis epidermal nevus can be divided into two types: keratinocytic or non organoidand organoid type.³ Keratinocytic epidermal nevus is the most common type of epidermal nevus. It occurs due to overgrowth of keratinocytes. Different varients of keratinocytic epidermal nevus are seen, such as linear epidermal nevus, hard nevus of Unna, soft epidermal nevus and nevus verrucosus etc.¹ On the other hand organoid type shows predominantly another component of skin.⁴ Epidermal nevus occurs as a result of activated genetic mutation in FGFR-3, HRAS or PIK3CA genes. FOXN1 is highly expressed in these lesions.¹ Most common pattern of keratinocytic nevus is linear epidermal nevus. The lesions are verrucous, skin-coloured dirty gray or brown coloured papule, which coalesce to form serpiginious plaques. They follow the line of Blaschko. These lines are thought to be representative pathways of epidermal cell migration and proliferation during development of fetus.⁵

Linear epidermal nevus may be either localized or systematized. In localized type, only one linear lesion is present and lesion is confined to one side of the body. Common sites are head, trunk and extremities. In systematized type there are many parallel linear lesions are seen. They may be unilateral or bilateral.

Localized and more commonly systematized linear epidermal nevus may be associated with skeletal deformity and CNS deficiency.⁶ Rarely squamous cell carcinoma or basal cell carcinoma may arise in epidermal nevus.⁷

Epidermal nevus may occur as a part of epidermal nevus syndrome and may be associated with internal manifestation. These syndromes have characteristic cutaneous findings and at times relevantly specific internal findings.¹  The six different types of epidermal nevus syndromes are nevus sebaceous, CHILD (congenital hemidysplasia with ichthyosiform erythroderma and limb defects) syndrome, nevus comedonicus, Becker’s nevus, Proteus syndrome, phacomatosispigmentokeratotica.

Histologically epidermis of epidermal nevus is hyperplastic. There is variable hyperkeratosis, papillomatosis and acanthosis with elongation of rete ridges. Upto 62% of biopsy specimens have these pattern and these are called non-epidermolytic epidermal nevus. About 16% of epidermal nevi show features of epidermolytic hyperkeratosis. Other histologic patterns are psoriatic type, acrokeratosisverruciformis like type and a Darier’s disease like type.¹ Epidermolytic hyperkeratosis is more common in systematized type than localized type. This reaction pattern of skin was first described by Ackerman in 1970.⁷It occurs due to defective keratin genes (KRT-1 and KRT-2), which causes excessive and abnormal keratinization. The salient histologic features are- compact hyperkeratosis, perinuclear vacuolization of the cells in stratum malpighii, irregular cellular boundaries and increased numbers of large irregular keratohyaline granules. Epidermolytic hyperkeratosis is seen in other conditions, such as- bullous congenital icthyosiformerythroderma, icthyosisbullosa of Siemens, Vorner’spalmoplanterkeratoderma, melanocytic nevus, epidermolyticacanthoma, basal cell carcinoma and squamous cell carcinoma etc.

Main clinical differential diagnosis of epidermal nevus areepidermodysplasiaverruciformis, inflammatory linear verrucous epidermal nevus, linear psoriasis and lichenstriatus.Epidermodysplasiaverruciformis is a genetic disease characterized by HPV infection. This is usually associated with HPV 5 or 8, less commonly 3, 4, 5.¹ Histologically this lesion is characterized by acanthosis, hyperkeratosis, large cells with blue-gray cytoplasm, often with dysplastic change and irregular granular layer with rare perinuclear halo. Inflammatory linear verrucous epidermal nevus (ILVEN) is a type of epidermal nevus. These lesions are also distributed in linear pattern, but they are erythematous and itchy. Histologically ILVEN is characterized by some specific features, which are absent in epidermal nevus. These are- areas of depressed orthokeratosis with underlyinghypergranulosis, alternating areas of slightly raised parakeratosis with underlying hypogranulosis.⁸ In linear psoriasis, the lesions may be pink to red papules or silvery scales. Typical histologic findings are regular elongation of rete ridges, thin suprapaillary plates, hypo or agranulosis, Munro microabcess and spongiform pustules of Kogoj, which are absent in epidermal nevus. In lichen striatus, the lesions are erythematous papules arranged in linear pattern following lines of Blaschko. But histologically it differs from epidermal nevus by presence of vacuolar alteration of basal layer and band like lymphocytic infiltrate.⁷ Treatment modalities of epidermal nevus are topical cream, cryotherapy, laser and dermabrasion. But management is difficult, because the lesions  recur unless treatment extend into dermis.

Conclusion

Epidermal nevus with epidermolytic hyperkeratosis is a rare condition. As this is a mosaic genetic disorder of suprabasal keratin, it can be transmitted to offspring and produce generalized epidermolytic hyperkeratosis. So genetic counseling is essential for these patients.

References

1. James W, Berger T, Elston D, Neuhaus I. Andrew’s Diseases of the Skin. 12th ed. Philadelphia: ELSEVIER. 2016. pp 625-26
2. Guite Z, Pamei D, Gunto H, Das K. Epidermolytic hyperkeratosis in verrucous epidermal nevus. Journal of medical society. 2014; 28(1): 47-8.
3. Pollozhani N, Damevska K, Silvija D, Adjievska N, Gocev G. Epidermolytic hyperkeratosis: clue for diagnosis. Global Dermatology.  2017; 4(1): 1-3.
4. Ngan V (2003). All about the skin. Retrieved from DermNet NZ website.  https://www.dermanet.org.nz
5. Kouzak SS, Mendes MS, Costa IM. Cutaneous mosaicisms: concepts, patterns and classifications. An Bras Dermatol. 2013;88(4):507-517.
6. Edler D. in Lever’s Histopathology of the Skin. 10^th Philadelphia: LIPPINCOTT WILLIAMS & WILKINS; 2009. pp: 791-92
7. Das A, Podder I, Das A, Ghosh A, Shome K. Epidermolyticblaschkoidverrucous epidermal nevus: Report of two cases. Indian J DermatopatholDiagnDermatol. 2015; 2:46-8.
8. Meibodi NT, Nahidi Y, Jaridi Z. Epidermolytic hyperkeratosis in inflammatory linear verrucous epidermal nevus. Indian J Dermatol. 2011; 56:309-12.

Papillary Thyroid Carcinoma Arising within Mature Ovarian Teratoma: A Case Report

Sadaf A, ¹ Hossain MI,²  Sultana  N,³ Nasreen S,⁴ Rahman Z⁵

1. *Dr. Anika Sadaf, MD. (Pathology) Phase B, Resident, Department of Pathology, Chittagong Medical College, Chattogram, Bangladesh, 4203. anikasadaf261189@gmail.com.
2. Mohammad Ismail Hossain. Lecturer, Department of Pathology, Chittagong Medical College, Chattogram, Bangladesh, 4203.
3. Nahid Sultana. Senior Consultant, Obstetrics & Gynaecology. 250 Bedded General Hospital,Chandpur, Bangladesh, 3600.
4. Sayeeda Nasreen. Assistant Professor, Department of Pathology, Chittagong Medical College, Chattogram, Bangladesh, 4203.
5. Zillur Rahman. Professor, Department of Pathology, Chittagong Medical College, Chattogram, Bangladesh, 4203.

 *For correspondence

 Abstract

Mature cystic teratoma is the commonest ovarian germ cell tumor. Though malignant transformation is uncommon, papillary thyroid carcinoma has rarely been described as associated with ovarian teratomas. We report a case of a 34-years old multiparous woman who presented with acute abdominal pain and an ovarian mass. After salphingo-oophorectomy, the patient was diagnosed as papillary thyroid carcinoma that arose within a mature cystic ovarian teratoma. To our knowledge, this is the first reported case of papillary thyroid carcinoma arising within a mature ovarian teratoma in this tertiary health care center in Chattogram. We recommend long term follow up to see any metastatic possibility.

 [Journal of Histopathology and Cytopathology, 2020 Jan; 4 (1):60-64]

Keywords: Mature cystic teratoma, papillary thyroid carcinoma, struma ovarii.

Introduction

Among the ovarian germ cell tumors Mature Cystic Teratoma (MCT) is the most common and comprises 10–20% of all ovarian tumors. However, malignant transformation of MCT is not common and the incidence is 1–3%.¹ Squamous Cell Carcinoma (SCC) is the commonest type, found in 80% of cases.² Papillary thyroid carcinoma (PTC) within teratoma is one of the rarest types with ranges varying from 0.1% and 0.2%, and usually diagnosed postoperatively.³ The synchronous development of malignant struma ovarii and primary thyroid carcinoma is extremely rare, though a handful of cases were reported.⁴ So, presence of thyroid tissue in teratoma, should proceed with further work up to confirm the diagnosis and to explore the possibility of a malignant lesion in the mass- either primary or metastasis.⁵ Here we present a case of a patient with a PTC arising within a ovarian MCT.

Case Presentation
34-year-old multiparous woman who had abdominal pain, distention and irregular menstrual bleeding for approximately for 6 months, presented to the emergency service of Chittagong Medical College Hospital in May 2019 with the complaints of a sharp pain in lower abdomen, with accompanying vomitting. There was no previous medical or surgical history.

On abdominal examination, a tender mass adjacent to the left side of the umbilicus were detected. Manual examination of the vagina revealed tenderness and mass in left adnexal region. Paps smear was done with no abnormality detected. Haemogram and biochemical test results were normal except a hemoglobin level of 9.2 gm/dl. CA-125 was within normal limit and β-HCG was normal. Ultrasonography of the lower abdomen revealed a complex solid cystic mass measuring 82x62x55 mm, with mixed echotexture, compatible with dermoid cyst.

At laparotomy, a cystic mass of approximately 9x7cm size, with a white, smooth glistening surface, originating from the left ovary was observed. Opposite ovary was apparently normal and no adhesion or intra-abdominal deposit was observed. Left sided salphingo-oophorectomy was performed preserving the uterus and right ovary and sent to Department of Pathology, Chittagong Medical College, Chattogram for histopathological evaluation. On gross pathological examination, a cystic mass of 9 cm in diameter (fig-1) with 3cm fallopian tube were noted. On cross section, hair, sebum & fatty materials were come out and some thick greenish fluid was drained and some solid structure was observed on its wall (fig-2).

Microscopic examination revealed mature teratomatous component represented by skin with associated adnexal structures, muscles, fat, benign glands lined by mucin containing columnar epithelium (fig-3,4) and thyroid tissue, within the thyroid tissue foci papillary thyroid carcinoma (fig-5,6) was found. Lining cells had oval nuclei showing nuclear overlapping, grooving and intranuclear cytoplasmic inclusions. Follicles also contained amorphous eosinophilic thick colloid. The fallopian tube was unremarkable. Immunohistochemical (IHC) examination revealed positivity for TTF-1 (fig-8,9). With these findings, diagnosis of a “mature cystic teratoma with malignant transformation to papillary thyroid carcinoma” was made. Postoperatively plasma levels of T₃, T₄, TSH and thyroglobulin of the patient were normal. A normal parenchymal vasculature was identified by postoperative ultrasonography of the thyroid gland.

Discussion

Mature cystic teratomas are also known as dermoid cysts, because they are mostly cystic, skin & skin appendages are the most common structures. Thyroid tissue is present in 10% of the all cases.⁶ Teratomas containing more than 50% of thyroid tissues are called struma ovarii, often presented as monodermal teratoma.⁷ Malignant transformation of MCT is rare; however, several types of malignancy can develop from any one of three germ-cell layers. Squamous cell carcinoma, derived from ectoderm is the commonest type; less common malignancies include soft tissue sarcomas, adenocarcinomas, malignant melanomas, basal cell carcinomas, carcinoid tumors, and thyroid carcinomas.² Among thyroid carcinomas the most common histological type is the papillary carcinoma (44%), other types are follicular carcinoma (30%) and follicular variant of papillary carcinoma (26%).³

 The malignant change of an initially benign cystic teratoma is detected in patients between 40 and 60 years of age, older than its benign counterpart. Although the cancer occurs at any age, most patients are postmenopausal.¹ The tumor may present as pelvic discomfort, with a pelvic mass on abdominal imaging (USG, CT, MRI) or during laparotomy for any other reason. Preoperative definitive diagnosis of stroma ovarii or papillary thyroid carcinoma is not possible. The only possibility of preoperative diagnosis is by radioactive iodine scan (not done routinely).⁸ Various case reports have been published over the past few years regarding the histological diagnoses and treatment options. The diagnoses of thyroid carcinomas arising in teratomas should be made following the guidelines for diagnosing carcinomas in thyroid gland. Disease is treatable with good out come in most cases. Only 7% and 14% of patients with papillary carcinoma and typical follicular carcinoma, respectively died of disease. Due to rarity of disease no consensus on treatment has been made, however treatment options include oophorectomy, additional thyroidectomy, radioactive iodine and long term follow up with serum thyroglobulin measurement.⁹

In order to determine metastatic disease, in MCT cases undergoing malignant transformation, follow up of thyroglobulin (Tg) levels is recommended. The only source of circulating Tg is the thyroid tissue and ovarian teratomas containing thyroid tissue, which is a very rare condition. However, high Tg level in benign thyroid diseases hamper determination of it as a convenient tumour marker in MCT, who did not undergo thyroidectomy and who contain thyroid tissue with malignant transformation. On the other hand, the high levels of anti-thyroglobulin antibody (anti-Tg Ab) may cause Tg levels to be erroneously low. For this reason, the follow-up of Tg levels is favourable for patients, who underwent thyroidectomy only and for patients left with no or very little thyroid tissue. In order to evaluate Tg levels correctly, follow-up of Tg levels together with anti-Tg Ab levels is advisable as persisting high levels of anti-Tg Ab indicate a persistent disease.³ In our case, Plasma T₃, T₄, TSH and Tg level were normal and normal parenchymal vasculature was identified by ultrasonography of the thyroid gland. Anti-Tg Ab level can’t be performed due to patient’s refusal. Logani et al., 2001 was commented the absence of normal thyroid tissue and features of teratoma, in favour of a metastatic lesion originating from thyroid gland.¹⁰ In the presented case, histologic evidence of mature teratoma, normal thyroid tissue along foci of papillary thyroid carcinoma, and positive immunohistochemical stain for thyroid transcription factor-1(TTF-1) indicates primary thyroid carcinoma arising within MCT.

Conclusion

Whether further therapy with total thyroidectomy and radioiodine ablation may be beneficial is unknown. The rarity of MCT cases undergoing PTC transformation impedes the establishment of a protocol for treatment and follow-up. We recommend that a long-term follow-up of these cases is needed to know more about the prognosis and to see any local recurrence or metastasis.

References

1. Rim SY, Kim SM, Choi HS. Malignant transformation of ovarian mature cystic teratoma. Int J Gynecol Cancer. 2006; 16:140–44.
2. Pineyro MM, Pereda J, Schou P, Santos DL, Peña SDI, Caserta B,Pisabarro R. Papillary thyroid microcarcinoma arising within a mature ovarian teratoma: case report and review of the literature. Clinical Medicine Insights: Endocrinology and Diabetes. 2017; 10: 1–3.
3. Cokmez H,Gulbahar A, Yigit S, Aydin C. Oncocytic and tall columnar type papillary thyroid carcinoma arising on a mature cystic teratoma: A case report and literature review. J Pak Med Assoc. 2019; 69:116-19.
4. Tzelepis EG, Barengolts E, Garzon S, Shulan J, Eisenberg Y. Unusual case of malignant strumaovarii and cervical thyroid cancer preceded by ovarian teratoma: case report and review of the literature. Hindawi: Case Reports in Endocrinology. 2019 Mar 17; 1-7.
5. Yeasmin S. A case of papillary thyroid cancer and extraovarian pelvic Teratoma.Journal of the Endocrine Society. 2019; 3(1). available at:https://doi.org/10.1210/js.2019-SUN-603.
6. Bedir R,Yılmaz R. Coexistence of papillary thyroid cancer and hashimoto’sthyroiditis developing within an ovarian mature cystic teratoma. Journal of Mid-life Health. 2019 April 10; 10: 45-47.
7. ParulskaES, Pioch A, Chyrek EC, Wolinski K, Jurczyszyn DJ, Jedynska MJ, Majewski P, Zabel M,Ruchala M. The role of immunohistochemical examination in diagnosis of papillary thyroid cancer in strumaovarii. Folia Histochemica Et Cytobiologica. 2019;57(1):35–42.
8. Naeem M, Iqbal M, Imran MB,Tabassum R. Malignant strumaovarii: a rare case report. European Journal of Medical Case Reports. 2017; 2(1): 30-32.
9. Haider A, Hussain M, Hassan U, Loya A. Papillary thyroid carcinoma arising in ovarian teratomas: A report of three cases. Journal of Islamabad Medical & Dental College (JIMDC). 2015 Sep 27; 4(2): 88-90.
10. Logani S, Baloch ZW, Snyder PJ, Weinstein R, LiVolsi VA. Cystic ovarian metastasis from papillary thyroid carcinoma: A case report. Mary Ann Liebert, Inc. 2001; 11(11): 1073-1075.