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Rare case of retroperitoneal liposarcoma presented as perineal swelling (The tip of Iceberg)

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Case Report

Rare case of retroperitoneal liposarcoma presented as perineal swelling (The tip of Iceberg)

  • Noha M. Taha 1*
  • Mohamed I. Ali 2
  • Mohamed Al-wazieery 3
  • Eldesoky Ibrahim Nouh 4
  • Ismail M Ali 5
  • Ayed Y. Asiri 6

*Corresponding Author: Noha Mohamed Taha, Radiology Department, Faculty of medicine, Ain shams university, Cairo, Egypt.

Citation: Noha M. Taha, Mohamed I. Ali, Mohamed Al-wazieery, Eldesoky Ibrahim Nouh, Ismail M Ali, et al, (2024), Rare case of retroperitoneal liposarcoma presented as perineal swelling (The tip of Iceberg), J. Cancer Research and Cellular Therapeutics, 8(5); DOI:10.31579/2640-1053/204

Copyright: © 2024, Noha Mohamed Taha, this is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.

Received: 23 July 2024 | Accepted: 05 August 2024 | Published: 12 August 2024

Keywords: retroperitoneal liposarcoma; mri; subcutaneous fat

Abstract

Background: Primary typical and atypical lipomatous tumors are the most common soft tissue tumors, where liposarcomas are the most common sarcoma. They are mostly found in the retroperitoneum and extremities. Rarely these tumors could present as hernias through different hernial orifices, this case report presents a rare presentation of retroperitoneal liposarcoma presented as a perineal swelling herniating through the ischiorectal and ischioanal fossa.

Case presentation: A 53 years old male patient presented to the outpatient surgery clinic in Al-Hayat National Hospital, Riyadh, kingdom of Saudi Arabia (KSA) complaining of swelling in the left gluteal area for two years, that progressively increased in size with no other symptoms. Magnetic resonance imaging (MRI) revealed a large retroperitoneal soft tissue mass lesion of fat content herniating into the perineal region. Complete surgical excision of this mass were done.

Conclusions: liposarcoma herniating through ischiorectal and ischioanal fossa are rare form of retroperitoneal liposarcoma as in this case where radiology plays an important role in initial diagnosis, staging and surgical planning.

Introduction

Retroperitoneal sarcomas (RPS) are aggressive and rare type of malignant neoplasm, with incidence of about 0.5–1/100,000 inhabitants per year and account for 10–16% of all sarcomas [1]. RPS include heterogeneous and complex group of neoplasia, with four types represent almost 90% of all cases, these are, liposarcomas (LPS), leiomyosarcomas, solitary fibrous tumor, and malignant peripheral nerve sheath tumor [2].

Liposarcomas are the most common type of soft tissue sarcomas, they predominantly occur in the retroperitoneum and thigh [3]. Liposarcomas accounts for 20% of all retroperitoneal tumors and has multiple subtypes, including atypical, well-differentiated and dedifferentiated liposarcomas [4]. Due to the uncontrolled growth in the large potential space of the retroperitoneum, they mostly present with symptoms of mass effect with large median size reaching around 30 cm at the time of presentation making surgical management challenging [5]. Due to the large size of lipomatous tumors, they sometimes present as hernias which can occur through different hernial orifices either in the abdominal wall or in the groin. The herniated part of the tumor represents only the ‘tip of the iceberg’, as the main part of the tumor is not visualized clinically and is often underestimated, also this late presentation results in higher risk of de-differentiation [6].

Case Report

A 53 years-old male presented to the outpatient surgery clinic in Al-Hayat National Hospital, Riyadh, KSA complaining of a swelling in the left gluteal area for two years (figure. 1), that progressively increased in size with no other symptoms. On clinical examination, the swelling was huge in size, soft, not tender not hot with normal overlying skin.

On clinical examination a huge, not tender, pedunculated mass was seen.

Figure 1: photo of the swelling on clinical exam

Ultrasound (US) examination was suspicious for a large gluteal lipoma. The surgeon requested MRI study with contrast for better assessment of the swelling and for pre-operative planning. 

MRI was done including axial T1, T2, STIR WIs, coronal T2 and sagittal T2 WIs, then contrast was injection and axial, coronal and sagittal T1 fat suppressed WIs were acquired. On pre contrast images a large well-defined dumbbell shaped mass measuring 11.2 x 9.8 x 31 cm in the AP, TR, and CC dimensions respectively, was seen in the retroperitoneum extending to the left perineal region, filling the lower retroperitoneal/presacral space markedly displacing and compressing the 

urinary bladder (UB) and prostate anteriorly (fig. 2a), inferiorly it was seen extending downwards at the left ischiorectal fossa, markedly compressing and displacing the rectum to the right side, also this lesion was seen herniating through the left pelvic floor muscles (left levator ani muscle) into the left ischio-anal fossa, displacing the anal canal to the right side and protruding outwards in the perineum (figure 2b). This lesion displayed predominantly hyperintense signal on T1 and T2 WIs that was suppressed in most of the lesion on STIR WIs (figure 3a, b, c). On post contrast images it showed heterogenous enhancing soft tissue components and thick internal septations (figure 4).

Figure 2: MRI examination of the mass

(A) Sagittal T2WIs shows a large dumbell shaped soft tissue lesion seen filling the presacral space and extending downwards into the perineal region. The prostate is seen markedly displaced and compressed anteriorly (red arrow). (B) Coronal T2 WIs revealed large lesion een extending from the retroperitoneal region inferiorly in the left ischiorectal fossa then through the left pelvic floor muscles (left levator ani muscle) downwards 

in the left ischio-anal fossa, to protrude through the perineum.(C) Coronal T1 WIs (D) coronal T2WIs, (E) STIR WIs revealed large soft tissue lesion displaying hyperintense signal in T1 and T2 WIs, most of the lesion is seen suppressed in STIR WIs with internal hyperintense soft tissue components and internal septations. (F)Post contrast MRI images of the lesion: Sagittal T1 fat suppressed post contrast images showed internal heterogeneously enhancing soft tissue components and thick enhancing septations.

Figure 3: The mass post-surgical excision

Figure 4: The histopathology of the excised maas show:

A) well-circumscribed hypocellular tumor composed of mature fat plus variably sized adipocytes and fibro-myxoid spindle cells. B) fibrous tissue septae are present containing spindle cells and highly pleomorphic cells, some nuclei have sharply outlined vacuoles (Lochkem) with very few lipoblasts.

The patient was prepared for surgery. On pre-operative assessment, according to The American Society of Anesthesiologists (ASA) classification the patient was ASA1, Exploration laparotomy was through midline incision, the small bowel loops were retracted upwards, the sigmoid colon and rectum were pushed to the right side. Opening of the posterior parietal peritoneum was done and the mass was localized and dissected after securing both ureters laterally. The herniated part was pushed up into the pelvis through retroperitoneal blunt dissection until the mass was completely excised. Hemostasis and then close of the peritoneal defect were done, intra-abdominal suction drain was inserted then the incision was closed in layers (figure 5).

Transversus abdominus Plane (TAP) block was given at the end of operation and before awaking the patient for post-operative pain relief. Reverse given to awake the patient by Sugammadex. No mishaps during the time of anesthesia. After operation the patient was shifted to recovery room for 30 minutes, then admitted to ICU for monitoring of the patient vital signs as he had major surgery and to check his CBC, PH level, ABG and he spent 24 hours there. Histopathology of the excised mass confirmed the diagnosed of well differentiated liposarcoma (fig 6). The patient came for follow up in the surgery clinic which revealed complete disappearance of the large perineal mass. MRI assessment was done one month after surgery which revealed post-operative seroma with no sizable residual tumoral tissue

Discussion

The current case represents one of the rare tumours - giant retroperitoneal well-differentiated liposarcoma that measured 30 cm in the maximal dimension -. Most retroperitoneal liposarcomas are large at presentation; nearly 50% of retroperitoneal sarcomas are greater than 20 cm at diagnosis [7]. Obstructive complications and organ displacement are the most prevalent symptoms that they manifest later [8]. According to the WHO 2020 classification, there are five main subtypes of liposarcomas: atypical lipomatous tumor (ALT)/well-differentiated liposarcoma (WDLPS), dedifferentiated, myxoid, pleomorphic, and myxoid pleomorphic [9]. All subtype of liposarcoma develops in the retroperitoneum. Most of them are dedifferentiated subtypes and well-differentiated liposarcomas. In the retroperitoneum, pleomorphic and myxoid liposarcomas are uncommon. Well-differentiated liposarcoma (ALT/WDLPS) is a mesenchymal tumor that is locally aggressive but does not spread. It is mostly composed of adipocytes and stromal cells, and it has focal nuclear atypia in both of these cell types [10]. Approximately 40–45% of all liposarcomas are ALT/WDLPS. The deep soft tissue of the trunk and proximal extremities is where it most usually manifests. Moreover, the retroperitoneum is frequently affected as in the present case. WDLPS is a synonym for ALT. Tumor location and resectability are the main factors that determine whether to use either term [10]. Retroperitoneal tumors are extremely difficult to completely excise and are best categorized as WDLPS. Three subtypes of ALT/WDLPS are distinguished histologically: (1) adipocytic (lipoma-like), (2) sclerosing, and (3) inflammatory [11]. The presence of more than one morphological pattern in the same lesion is common, particularly in retroperitoneal tumors. [12]. Dedifferentiated liposarcoma (DDLPS) is an ALT/WDLPS that progresses to a varied histological grade of sarcoma, typically non-lipogenic, either in the primary or recurrent phase [13]. A well-differentiated component may not be found. The high-grade component could sporadically be lipogenic. Roughly 90% of cases arise de novo, while 10

Conclusions

Retroperitoneal liposarcomas are rare soft tissue tumors, Familiarity with different imaging findings of liposarcomas and its rare presentations as a hernia through different hernial orifices is critical to ensure accurate preoperative diagnosis as well as proper treatment and surgical planning.

List of abbreviations

(KSA)  kingdom of Saudi Arabia,  (MRI) Magnetic resonance imaging, (CT) computerized tomography, (US) ultrasound, T1 time 1, T2 time 2, (STIR) short tau inversion recovery, (ASA) American Society of Anesthesiologists, (TAP) Transversus abdominus  plane, (ALT/ WDLPS)) atypical lipomatous tumor/well-differentiated liposarcoma, Dedifferentiated liposarcoma (DDLPS), pleomorphic liposarcoma (PLPS), Myxoid liposarcoma (MLPS), malignant peripheral nerve sheath tumors (MPNSTs), solitary fibrous tumor (SFT), Arterial blood gases (ABG), Anteroposterior (AP), Complete blood count (CBC), craniocaudal (CC), Liposarcoma (LPS), Transverse (Tr). Retroperitoneal sarcoma (RPS). Weighted images (WIs).

Declarations

Ethics approval and consent to participate:

 This study was approved by the Research Ethics Committee of the Hayat National Hospitals, the requirement for written consent was waived in this case report and discussed and approved by the ethical committee.

Consent for publication: 

Not applicable.

Availability of data and materials:

The datasets used and/or analyzed during the current study are available from the corresponding author on reasonable request.

Competing interests:

The authors declare that they have no competing interest.

Funding:

No Funds, sponsorship or financial support to be disclosed

Authors contributions:

  • Noha M. Taha: Idea of the paper, diagnosis of the case in radiology department, attended the operation to describe exactly the pathway of the lesion and its relations, writing the manuscript. 
  • Ismail M Ali: writing and revising the manuscript.
  • Mohamed I. Ali: The surgeon who make the clinical examination and who did the operation.
  • Eldesoky Ibrahim Nouh: The anesthesia doctor who prepared the patient for operation and who made the anesthesia during the operation in the OR.
  • Ayed Y. Asiri: The ICU doctor who monitored the patient after this major operation.
  • All authors read and approved the final manuscript.

- Acknowledgements:

For the collaborative group including:

  • Mohamed Al-Wazieery: The second surgeon in the operation, helps in writing the manuscript as well as following the pathology report.
  • Dr. Mohamed S. Elshal. Registrar of Anesthesia at Benha teaching hospital and Hayat National Hospital, who helped in preoperative preparation of the patient as well as in the OR.
  • Mr. Louie Salmo. The excellent MRI technician who did the MRI study under the supervision of Dr. Noha M. Taha.

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International Journal of Clinical Case Reports and Reviews. I strongly recommend to consider submitting your work to this high-quality journal. The support and availability of the Editorial staff is outstanding and the review process was both efficient and rigorous.

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Andreas Filippaios

Thank you very much for publishing my Research Article titled “Comparing Treatment Outcome Of Allergic Rhinitis Patients After Using Fluticasone Nasal Spray And Nasal Douching" in the Journal of Clinical Otorhinolaryngology. As Medical Professionals we are immensely benefited from study of various informative Articles and Papers published in this high quality Journal. I look forward to enriching my knowledge by regular study of the Journal and contribute my future work in the field of ENT through the Journal for use by the medical fraternity. The support from the Editorial office was excellent and very prompt. I also welcome the comments received from the readers of my Research Article.

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Dr Suramya Dhamija

Dear Erica Kelsey, Editorial Coordinator of Cancer Research and Cellular Therapeutics Our team is very satisfied with the processing of our paper by your journal. That was fast, efficient, rigorous, but without unnecessary complications. We appreciated the very short time between the submission of the paper and its publication on line on your site.

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Bruno Chauffert

I am very glad to say that the peer review process is very successful and fast and support from the Editorial Office. Therefore, I would like to continue our scientific relationship for a long time. And I especially thank you for your kindly attention towards my article. Have a good day!

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Baheci Selen

"We recently published an article entitled “Influence of beta-Cyclodextrins upon the Degradation of Carbofuran Derivatives under Alkaline Conditions" in the Journal of “Pesticides and Biofertilizers” to show that the cyclodextrins protect the carbamates increasing their half-life time in the presence of basic conditions This will be very helpful to understand carbofuran behaviour in the analytical, agro-environmental and food areas. We greatly appreciated the interaction with the editor and the editorial team; we were particularly well accompanied during the course of the revision process, since all various steps towards publication were short and without delay".

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Jesus Simal-Gandara

I would like to express my gratitude towards you process of article review and submission. I found this to be very fair and expedient. Your follow up has been excellent. I have many publications in national and international journal and your process has been one of the best so far. Keep up the great work.

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Douglas Miyazaki

We are grateful for this opportunity to provide a glowing recommendation to the Journal of Psychiatry and Psychotherapy. We found that the editorial team were very supportive, helpful, kept us abreast of timelines and over all very professional in nature. The peer review process was rigorous, efficient and constructive that really enhanced our article submission. The experience with this journal remains one of our best ever and we look forward to providing future submissions in the near future.

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Dr Griffith

I am very pleased to serve as EBM of the journal, I hope many years of my experience in stem cells can help the journal from one way or another. As we know, stem cells hold great potential for regenerative medicine, which are mostly used to promote the repair response of diseased, dysfunctional or injured tissue using stem cells or their derivatives. I think Stem Cell Research and Therapeutics International is a great platform to publish and share the understanding towards the biology and translational or clinical application of stem cells.

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Dr Tong Ming Liu

I would like to give my testimony in the support I have got by the peer review process and to support the editorial office where they were of asset to support young author like me to be encouraged to publish their work in your respected journal and globalize and share knowledge across the globe. I really give my great gratitude to your journal and the peer review including the editorial office.

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Husain Taha Radhi

I am delighted to publish our manuscript entitled "A Perspective on Cocaine Induced Stroke - Its Mechanisms and Management" in the Journal of Neuroscience and Neurological Surgery. The peer review process, support from the editorial office, and quality of the journal are excellent. The manuscripts published are of high quality and of excellent scientific value. I recommend this journal very much to colleagues.

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S Munshi

Dr.Tania Muñoz, My experience as researcher and author of a review article in The Journal Clinical Cardiology and Interventions has been very enriching and stimulating. The editorial team is excellent, performs its work with absolute responsibility and delivery. They are proactive, dynamic and receptive to all proposals. Supporting at all times the vast universe of authors who choose them as an option for publication. The team of review specialists, members of the editorial board, are brilliant professionals, with remarkable performance in medical research and scientific methodology. Together they form a frontline team that consolidates the JCCI as a magnificent option for the publication and review of high-level medical articles and broad collective interest. I am honored to be able to share my review article and open to receive all your comments.

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Tania Munoz

“The peer review process of JPMHC is quick and effective. Authors are benefited by good and professional reviewers with huge experience in the field of psychology and mental health. The support from the editorial office is very professional. People to contact to are friendly and happy to help and assist any query authors might have. Quality of the Journal is scientific and publishes ground-breaking research on mental health that is useful for other professionals in the field”.

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George Varvatsoulias

Dear editorial department: On behalf of our team, I hereby certify the reliability and superiority of the International Journal of Clinical Case Reports and Reviews in the peer review process, editorial support, and journal quality. Firstly, the peer review process of the International Journal of Clinical Case Reports and Reviews is rigorous, fair, transparent, fast, and of high quality. The editorial department invites experts from relevant fields as anonymous reviewers to review all submitted manuscripts. These experts have rich academic backgrounds and experience, and can accurately evaluate the academic quality, originality, and suitability of manuscripts. The editorial department is committed to ensuring the rigor of the peer review process, while also making every effort to ensure a fast review cycle to meet the needs of authors and the academic community. Secondly, the editorial team of the International Journal of Clinical Case Reports and Reviews is composed of a group of senior scholars and professionals with rich experience and professional knowledge in related fields. The editorial department is committed to assisting authors in improving their manuscripts, ensuring their academic accuracy, clarity, and completeness. Editors actively collaborate with authors, providing useful suggestions and feedback to promote the improvement and development of the manuscript. We believe that the support of the editorial department is one of the key factors in ensuring the quality of the journal. Finally, the International Journal of Clinical Case Reports and Reviews is renowned for its high- quality articles and strict academic standards. The editorial department is committed to publishing innovative and academically valuable research results to promote the development and progress of related fields. The International Journal of Clinical Case Reports and Reviews is reasonably priced and ensures excellent service and quality ratio, allowing authors to obtain high-level academic publishing opportunities in an affordable manner. I hereby solemnly declare that the International Journal of Clinical Case Reports and Reviews has a high level of credibility and superiority in terms of peer review process, editorial support, reasonable fees, and journal quality. Sincerely, Rui Tao.

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Rui Tao

Clinical Cardiology and Cardiovascular Interventions I testity the covering of the peer review process, support from the editorial office, and quality of the journal.

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Khurram Arshad

Clinical Cardiology and Cardiovascular Interventions, we deeply appreciate the interest shown in our work and its publication. It has been a true pleasure to collaborate with you. The peer review process, as well as the support provided by the editorial office, have been exceptional, and the quality of the journal is very high, which was a determining factor in our decision to publish with you.

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Gomez Barriga Maria Dolores

The peer reviewers process is quick and effective, the supports from editorial office is excellent, the quality of journal is high. I would like to collabroate with Internatioanl journal of Clinical Case Reports and Reviews journal clinically in the future time.

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Lin Shaw Chin

Clinical Cardiology and Cardiovascular Interventions, I would like to express my sincerest gratitude for the trust placed in our team for the publication in your journal. It has been a true pleasure to collaborate with you on this project. I am pleased to inform you that both the peer review process and the attention from the editorial coordination have been excellent. Your team has worked with dedication and professionalism to ensure that your publication meets the highest standards of quality. We are confident that this collaboration will result in mutual success, and we are eager to see the fruits of this shared effort.

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Maria Dolores Gomez Barriga

Dear Dr. Jessica Magne, Editorial Coordinator 0f Clinical Cardiology and Cardiovascular Interventions, I hope this message finds you well. I want to express my utmost gratitude for your excellent work and for the dedication and speed in the publication process of my article titled "Navigating Innovation: Qualitative Insights on Using Technology for Health Education in Acute Coronary Syndrome Patients." I am very satisfied with the peer review process, the support from the editorial office, and the quality of the journal. I hope we can maintain our scientific relationship in the long term.

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Dr Maria Dolores Gomez Barriga

Dear Monica Gissare, - Editorial Coordinator of Nutrition and Food Processing. ¨My testimony with you is truly professional, with a positive response regarding the follow-up of the article and its review, you took into account my qualities and the importance of the topic¨.

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Dr Maria Regina Penchyna Nieto

Dear Dr. Jessica Magne, Editorial Coordinator 0f Clinical Cardiology and Cardiovascular Interventions, The review process for the article “The Handling of Anti-aggregants and Anticoagulants in the Oncologic Heart Patient Submitted to Surgery” was extremely rigorous and detailed. From the initial submission to the final acceptance, the editorial team at the “Journal of Clinical Cardiology and Cardiovascular Interventions” demonstrated a high level of professionalism and dedication. The reviewers provided constructive and detailed feedback, which was essential for improving the quality of our work. Communication was always clear and efficient, ensuring that all our questions were promptly addressed. The quality of the “Journal of Clinical Cardiology and Cardiovascular Interventions” is undeniable. It is a peer-reviewed, open-access publication dedicated exclusively to disseminating high-quality research in the field of clinical cardiology and cardiovascular interventions. The journal's impact factor is currently under evaluation, and it is indexed in reputable databases, which further reinforces its credibility and relevance in the scientific field. I highly recommend this journal to researchers looking for a reputable platform to publish their studies.

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Dr Marcelo Flavio Gomes Jardim Filho

Dear Editorial Coordinator of the Journal of Nutrition and Food Processing! "I would like to thank the Journal of Nutrition and Food Processing for including and publishing my article. The peer review process was very quick, movement and precise. The Editorial Board has done an extremely conscientious job with much help, valuable comments and advices. I find the journal very valuable from a professional point of view, thank you very much for allowing me to be part of it and I would like to participate in the future!”

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Zsuzsanna Bene

Dealing with The Journal of Neurology and Neurological Surgery was very smooth and comprehensive. The office staff took time to address my needs and the response from editors and the office was prompt and fair. I certainly hope to publish with this journal again.Their professionalism is apparent and more than satisfactory. Susan Weiner

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Dr Susan Weiner

My Testimonial Covering as fellowing: Lin-Show Chin. The peer reviewers process is quick and effective, the supports from editorial office is excellent, the quality of journal is high. I would like to collabroate with Internatioanl journal of Clinical Case Reports and Reviews.

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Lin-Show Chin

My experience publishing in Psychology and Mental Health Care was exceptional. The peer review process was rigorous and constructive, with reviewers providing valuable insights that helped enhance the quality of our work. The editorial team was highly supportive and responsive, making the submission process smooth and efficient. The journal's commitment to high standards and academic rigor makes it a respected platform for quality research. I am grateful for the opportunity to publish in such a reputable journal.

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Sonila Qirko

My experience publishing in International Journal of Clinical Case Reports and Reviews was exceptional. I Come forth to Provide a Testimonial Covering the Peer Review Process and the editorial office for the Professional and Impartial Evaluation of the Manuscript.

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Luiz Sellmann