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Erschienen in: BMC Clinical Pathology 1/2016

Open Access 01.12.2016 | Case report

Uterine myometrial mature teratoma presenting as a uterine mass: a review of literature

verfasst von: Emmanuel Kamgobe, Anthony Massinde, Dismas Matovelo, Edgar Ndaboine, Peter Rambau, Tito Chaula

Erschienen in: BMC Clinical Pathology | Ausgabe 1/2016

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Abstract

Background

Teratomas are a germ cell tumors composed of two or more tissues which originate from ectoderm, endoderm or mesoderm. These tumors commonly arise from the ovary although other extragonadal sites can be involved, especially in children.

Case presentation

We report a case of a 21-year-old female of Sukuma ethnicity from the northern region of Tanzania who presented with abdominal pain and distension, fever, and abnormal vaginal discharge for the previous three weeks. The patient was also lactating for the previous 8 months following cesarean section delivery. Pelvic ultrasound suggested pelvic abscess but after laparotomy and histological analysis of a bulky uterus removed a diagnosis of mature uterine teratoma was confirmed.

Conclusion

Although it is rare, uterine teratoma should be considered in differential diagnosis to any patient with uterine mass even without typical radiological findings.
Hinweise

Competing interests

We report no conflict of interest and no funding was received for this work.

Authors’ contributions

EK reviewed the patient at emergency unit, did the surgery and drafted the first manuscript. AM reviewed manuscript drafts. DM drafted the first manuscript and reviewed manuscript drafts. EN reviewed manuscript drafts. PR read the histological slides and reviewed manuscript drafts. TC assisted the surgery and reviewed the manuscript which was later approved by all authors.
Abkürzungen
BMC
Bugando Medical Centre
H&E
Hematoxylin & Eosin

Background

Teratomas are usually composed of two or more embryonic germ layers: ectoderm, endoderm, and mesoderm. Extragonadal teratomas are ectopic to the location in which they are found. Teratomas can be classified as mature or immature on the basis of the presence or absence of immature neuroectodermal tissues in the tumor. Mature tumors have far less tendency to develop into malignancy compared to immature tumors. Teratomas are the most common gonadal tumors. They usually arise in the gonads and often occur in infancy and childhood. Extragonadal teratomas are rare and commonly develop in midline structures [1]. Uterine teratomas, which are part of extra gonadal teratomas of midline structures, account for 1–2 % of all teratomas. Complications during their surgical removal may occur depending on their location or if they are attached to any other structures. There is no documented evidence of metastatic potential [2, 3].
Primary teratomas of the uterus have rarely been reported since Mann’s first description of this entity in 1929 [1, 2]. Here we report a case of uterine mature teratoma in a 21-year-old woman with an exceptional presentation of this tumor.

Case presentation

A 21-year-old female of Sukuma ethnicity from the northern region of Tanzania presented at Bugando Medical Centre (BMC) outpatient clinic in Mwanza city with complaints of abdominal distension and pain, fever and abnormal vaginal discharge for the past 3 weeks. She was apparently lactating for the previous 8 months after cesarean section delivery of her first child. On physical examination, she appeared to be weak, febrile of about 38.5 °C with blood pressure of 110/70 mmHg. She was a blood group ‘A’ rhesus positive and her hemoglobin level was 6.3 g/dl.
On abdominal examination, a sub-umbilical midline scar was seen, and palpable suprapubic mass of about 16 weeks size of the uterus. The mass was soft, tender and mobile. The digital pelvic and vaginal examination elicited a closed cervix with tenderness on mobility and a non-bulging posterior fornix. There was no adnexal mass detected and gloved finger stained with pus-like discharge. Pelvic gynecological ultrasound suggested pelvic abscess (Fig. 1).
Patient was counseled for emergency laparotomy. Intraoperatively, the uterus was found to be bulky with discharging sinus on left fundal position. Both ovaries were healthy-looking and there was no fluid in the pouch of Douglas. The transverse incision was made on the uterus at the level of the discharging sinus. The yellowish mucinous tenacious materials with hairy tissues were observed. The decision to perform a total hysterectomy was reached; in which the removed uterus had hairs and sticky sebaceous matter found freely in the cavity. After surgery, the patient was transfused one unit of blood and intravenous antibiotics ceftriaxone, Gentamycin and Metronidazole were given with an addition of prophylactic Heparin. The patient had an uneventful recovery.
The sample was sent for histological examination. At the pathology department, the bisected uterus of 18 cm × 9 cm × 4 cm with no adnexa was identified. There was a cystic mass of 10 cm on the left fundal position in the myometrium containing hairs, sebaceous material, and pus.
The tissue sample was selected and sections were stained by Hematoxylin and Eosin (H&E) and observed by a light microscopy. Histology revealed a cyst in a myometrium contained keratin debris, and it was lined by squamous epithelium with dermal skin appendages with areas of denudation with lymphocyte and neutrophil infiltrate. The myometrium and endometrium was normal. Diagnosis of infected mature teratoma (dermoid cyst) was made (Figs. 2 and 3).
The patient has not shown any sign of disease recurrence for 8 months following hospital discharge.

Discussion

Mature cystic teratomas of uterus corpus are very rare and thought to originate from displaced germinal cells derived from pluripotential stem cells. The most commonly occurring mature cystic teratomas of the uterus are of the cervical part [2]. The presentation discussed here is unusual because there are only few reported cases of uterine teratomas to date arising from uterine body, and infected teratoma is also a rare event [2]. Extragonadal germ teratomas are very rare, accounting to 1–2 % of all teratomas [4, 5]. Since only few cases have been reported; we report the clinical presentation and pathological characteristics of patients with primary uterine mature teratoma in English literature (Table 1) [48].
Table 1
Clinical and pathological characteristics of patients with primary uterine mature teratoma in the literature
Author
Age
Symptoms
Site of Tumour
Histology
Treatment
Relapse
Treatment of Relapse
Our Case
21
Abdominal distension
Uterine corpus
Mature teratoma
Hysterectomy
None
 
Lim et al., [2011] [6]
27
Cervical polyp
Uterine cervix
Mature teratoma with some lymphoid elements
Excision
None
 
Newsom-Davis et al., [2009]
[5]
82
Postmenopausal bleeding
Uterine corpus
Mature& Immature Teratoma
Hysterectomy & bilateral salpingooophorectomy
Yes
Taxane, Etoposide & Cisplatin + Surgery
Cappelo et al., [2009] [4]
55
Asymptomatic (Multiple uterine leiomyomas)
Uterine corpus
Mature teratoma with thyroid differentiation
Hysterectomy
None
 
Wang et al., [2011] [7]
46
Abnormal uterine bleeding
Uterine corpus
Mature cystic teratoma
Hysterectomy
None
 
Papadia et al., [2007] [8]
58
Endometrial polyp/Abnormal uterine bleeding
Uterine corpus
Mature cystic teratoma
Excision
None
 
Our patient was in reproductive age, recently delivered and lactating; the presentation which is likely to develop teratoma due to the possibility of products of conceptus being implanted at any part along the reproductive tract during the process of fertilization according to the Blastomere Theory [810]. Although it does not apply to the patient discussed here, it is also possible for teratoma to develop in a newborn by abnormal migratory pathway of primordial germ cells from fetal yolk sac endodermal to the gonadal ridge during early embryogenesis as explained by Parthenogenic theory [3, 5].
Ultrasound images of the patient suggested pelvic abscess, but an explorative laparotomy and histopathological analysis of the excised bulky uterus led to a diagnosis of teratoma. In centers with, CT and MRI, those imaging techniques could have led to a correct diagnosis because of their higher accuracy compared to the ultrasound imaging [5]. Again, our hospital do not have Colour Doppler ultrasound which is superior to other imaging modalities with highest positive predictive value of 9.6 compared to 7.6 for MRI and 3.6 for CT16 [11].
Due to its rarity of occurrence, there is no standard guideline for management of mature uterine teratoma. The few case reports, however, suggest that the best management is complete tumor excision or total abdominal hysterectomy [3, 4, 68].

Conclusion

Although it is rare, uterine teratoma should be considered in differential diagnosis in any patient with uterine mass even without typical radiological findings.
"Written informed consent was obtained from the patient for publication of this Case report and any accompanying images. A copy of the written consent is available for review by the Editor of this journal".

Acknowledgment

The authors would like to thanks all the doctors and nurses at the BMC Emergency unit and Gynecological ward for taking good care of the patient and follow-up.
Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://​creativecommons.​org/​licenses/​by/​4.​0/​), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://​creativecommons.​org/​publicdomain/​zero/​1.​0/​) applies to the data made available in this article, unless otherwise stated.

Competing interests

We report no conflict of interest and no funding was received for this work.

Authors’ contributions

EK reviewed the patient at emergency unit, did the surgery and drafted the first manuscript. AM reviewed manuscript drafts. DM drafted the first manuscript and reviewed manuscript drafts. EN reviewed manuscript drafts. PR read the histological slides and reviewed manuscript drafts. TC assisted the surgery and reviewed the manuscript which was later approved by all authors.
Literatur
1.
Zurück zum Zitat Cortes J, Llompart M, Rossello J, Rifá J, Más J, Anglada P, et al. Immature teratoma primary of the uterine cervix. First case report. Eur J Gynaecol Oncol. 1989;11(1):37–42. Cortes J, Llompart M, Rossello J, Rifá J, Más J, Anglada P, et al. Immature teratoma primary of the uterine cervix. First case report. Eur J Gynaecol Oncol. 1989;11(1):37–42.
2.
Zurück zum Zitat Ben Ameur El Youbi M, Mohtaram A, Kharmoum J, Aaribi I, Kharmoum S, Bouzoubaa A, et al. Primary immature teratoma of the uterus relapsing as malignant neuroepithelioma: case report and review of the literature. Case Rep Oncol Med. 2013;2013:971803.PubMedPubMedCentral Ben Ameur El Youbi M, Mohtaram A, Kharmoum J, Aaribi I, Kharmoum S, Bouzoubaa A, et al. Primary immature teratoma of the uterus relapsing as malignant neuroepithelioma: case report and review of the literature. Case Rep Oncol Med. 2013;2013:971803.PubMedPubMedCentral
4.
Zurück zum Zitat Cappello F, Barbato F, Tomasino RM. Mature teratoma of the uterine corpus with thyroid differentiation. Pathol Int. 2000;50(7):546–8.CrossRefPubMed Cappello F, Barbato F, Tomasino RM. Mature teratoma of the uterine corpus with thyroid differentiation. Pathol Int. 2000;50(7):546–8.CrossRefPubMed
5.
Zurück zum Zitat Newsom-Davis T, Poulter D, Gray R, Ameen M, Lindsay I, Papanikolaou K, et al. Case report: Malignant teratoma of the uterine corpus. BMC Cancer. 2009;9(1):195.CrossRefPubMedPubMedCentral Newsom-Davis T, Poulter D, Gray R, Ameen M, Lindsay I, Papanikolaou K, et al. Case report: Malignant teratoma of the uterine corpus. BMC Cancer. 2009;9(1):195.CrossRefPubMedPubMedCentral
6.
Zurück zum Zitat Sc L, Ys K, Lee Y, Lee M, Jy L. Mature teratoma of the uterine cervix with lymphoid hyperplasia. Pathol Int. 2003;53(5):327–31.CrossRef Sc L, Ys K, Lee Y, Lee M, Jy L. Mature teratoma of the uterine cervix with lymphoid hyperplasia. Pathol Int. 2003;53(5):327–31.CrossRef
7.
Zurück zum Zitat Wang W-C, Lee M-S, Ko J-L, Lai Y-C. Origin of uterine teratoma differs from that of ovarian teratoma: a case of uterine mature cystic teratoma. Int J Gynecol Pathol. 2011;30(6):544–8.CrossRefPubMed Wang W-C, Lee M-S, Ko J-L, Lai Y-C. Origin of uterine teratoma differs from that of ovarian teratoma: a case of uterine mature cystic teratoma. Int J Gynecol Pathol. 2011;30(6):544–8.CrossRefPubMed
8.
Zurück zum Zitat Papadia A, Rutigliani M, Gerbaldo D, Fulcheri E, Ragni N. Mature cystic teratoma of the uterus presenting as an endometrial polyp. Ultrasound Obstet Gynecol. 2007;29(4):477–8.CrossRefPubMed Papadia A, Rutigliani M, Gerbaldo D, Fulcheri E, Ragni N. Mature cystic teratoma of the uterus presenting as an endometrial polyp. Ultrasound Obstet Gynecol. 2007;29(4):477–8.CrossRefPubMed
9.
Zurück zum Zitat NEWTON CW, ABELL MR. Iatrogenic fetal implants. Obstet Gynecol. 1972;40(5):686–91.PubMed NEWTON CW, ABELL MR. Iatrogenic fetal implants. Obstet Gynecol. 1972;40(5):686–91.PubMed
10.
Zurück zum Zitat Tyagi S, Saxena K, Rizvi R, Langley F. Foetal remnants in the uterus and their relation to other uterine heterotopia. Histopathology. 1979;3(4):339–45.CrossRefPubMed Tyagi S, Saxena K, Rizvi R, Langley F. Foetal remnants in the uterus and their relation to other uterine heterotopia. Histopathology. 1979;3(4):339–45.CrossRefPubMed
11.
Zurück zum Zitat Graham L. ACOG releases guidelines on management of adnexal masses. Am Fam Physician. 2008;77(9):1320–3.PubMed Graham L. ACOG releases guidelines on management of adnexal masses. Am Fam Physician. 2008;77(9):1320–3.PubMed
Metadaten
Titel
Uterine myometrial mature teratoma presenting as a uterine mass: a review of literature
verfasst von
Emmanuel Kamgobe
Anthony Massinde
Dismas Matovelo
Edgar Ndaboine
Peter Rambau
Tito Chaula
Publikationsdatum
01.12.2016
Verlag
BioMed Central
Erschienen in
BMC Clinical Pathology / Ausgabe 1/2016
Elektronische ISSN: 1472-6890
DOI
https://doi.org/10.1186/s12907-016-0026-8

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