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1
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Brown IS, Sokolova A, Rosty C, Graham RP. Cystic lesions of the retrorectal space. Histopathology 2023; 82:232-241. [PMID: 35962741 DOI: 10.1111/his.14769] [Citation(s) in RCA: 3] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/29/2022] [Revised: 08/03/2022] [Accepted: 08/09/2022] [Indexed: 12/24/2022]
Abstract
Cysts of the retrorectal space comprise a heterogeneous group of rare lesions. Most develop from embryological remnants and include tailgut cysts, dermoid cysts, rectal duplication cysts, anal canal duplication cysts, sacrococcygeal teratomas and anterior meningocoele. Tailgut cyst is the most common cyst of developmental origin, usually presenting as a multilocular cystic mass with mucoid content and lined by multiple epithelial types. Compared with tailgut cysts, rectal duplication cysts display all layers of the large bowel wall including a well-defined muscularis propria. Retrorectal cysts of non-developmental origin are far less common and represent lesions that either infrequently involve the retrorectal space or undergo extensive cystic change. This review provides an overview of the various histological types of cystic lesions of the retrorectal space, divided into cysts of developmental origin and those of non-developmental origin. A practical pathological and multidisciplinary approach to diagnosing these lesions is presented.
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Affiliation(s)
- Ian S Brown
- Envoi Specialist Pathologists, Brisbane, Queensland, Australia.,Royal Brisbane and Women's Hospital, Brisbane, Queensland, Australia.,University of Queensland, Brisbane, Queensland, Australia
| | - Anna Sokolova
- Sullivan Nicolaides Pathology, Brisbane, Queensland, Australia
| | - Christophe Rosty
- Envoi Specialist Pathologists, Brisbane, Queensland, Australia.,University of Queensland, Brisbane, Queensland, Australia
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2
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Wang YS, Guo QY, Zheng FH, Huang ZW, Yan JL, Fan FX, Liu T, Ji SX, Zhao XF, Zheng YX. Retrorectal mucinous adenocarcinoma arising from a tailgut cyst: A case report and review of literature. World J Gastrointest Surg 2022; 14:1072-1081. [PMID: 36185552 PMCID: PMC9521465 DOI: 10.4240/wjgs.v14.i9.1072] [Citation(s) in RCA: 3] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/23/2022] [Revised: 06/30/2022] [Accepted: 08/21/2022] [Indexed: 02/07/2023] Open
Abstract
BACKGROUND Tailgut cysts are defined as congenital cysts that develop in the rectosacral space from the residue of the primitive tail. As a congenital disease, caudal cysts are very rare, and their canceration is even rarer, which makes the disease prone to misdiagnosis and delayed treatment. We describe a case of caudal cyst with adenocarcinogenesis and summarize in detail the characteristics of cases with analytical value reported since 1990.
CASE SUMMARY A 35-year-old woman found a mass in her lower abdomen 2 mo ago. She was asymptomatic at that time and was not treated because of the coronavirus disease 2019 pandemic. Two weeks ago, the patient developed abdominal distension and right waist discomfort and came to our hospital. Except for the high level of serum carcinoembryonic antigen, the medical history and laboratory tests were not remarkable. Magnetic resonance imaging showed a well-defined, slightly lobulated cystic-solid mass with a straight diameter of approximately 10 cm × 9 cm in the presacral space, slightly high signal intensity on T2-weighted imaging, and moderate signal intensity on T1-weighted imaging. The mass was completely removed by laparoscopic surgery. Histopathological examination showed that the lesion was an intestinal mucinous adenocarcinoma, and the multidisciplinary team decided to implement postoperative chemotherapy. The patient recovered well, the tumor marker levels returned to normal, and tumor-free survival has been achieved thus far.
CONCLUSION The case and literature summary can help clinicians and researchers develop appropriate examination and therapeutic methods for diagnosis and treatment of this rare disease.
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Affiliation(s)
- Yan-Shuai Wang
- Department of General Surgery, The Fourth Affiliated Hospital, School of Medicine, Zhejiang University, Yiwu 322000, Zhejiang Province, China
| | - Qing-Yun Guo
- Department of Gynecology, Women's Hospital, School of Medicine, Zhejiang University, Hangzhou 310006, Zhejiang Province, China
| | - Fang-Hong Zheng
- Department of General Surgery, The Fourth Affiliated Hospital, School of Medicine, Zhejiang University, Yiwu 322000, Zhejiang Province, China
| | - Zi-Wei Huang
- Department of General Surgery, The Fourth Affiliated Hospital, School of Medicine, Zhejiang University, Yiwu 322000, Zhejiang Province, China
| | - Jia-Lang Yan
- Department of General Surgery, The Fourth Affiliated Hospital, School of Medicine, Zhejiang University, Yiwu 322000, Zhejiang Province, China
| | - Fu-Xiang Fan
- Department of General Surgery, The Fourth Affiliated Hospital, School of Medicine, Zhejiang University, Yiwu 322000, Zhejiang Province, China
| | - Tian Liu
- Department of Intensive Care Unit, The Fourth Affiliated Hospital, School of Medicine, Zhejiang University, Yiwu 322000, Zhejiang Province, China
| | - Shun-Xian Ji
- Department of Pathology, The Fourth Affiliated Hospital, School of Medicine, Zhejiang University, Yiwu 322000, Zhejiang Province, China
| | - Xiao-Feng Zhao
- Department of Gynecology, Women's Hospital, School of Medicine, Zhejiang University, Hangzhou 310006, Zhejiang Province, China
| | - Yi-Xiong Zheng
- Department of General Surgery, The Fourth Affiliated Hospital, School of Medicine, Zhejiang University, Yiwu 322000, Zhejiang Province, China
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3
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Martins P, Canotilho R, Peyroteo M, Afonso M, Moreira A, de Sousa A. Tailgut cyst adenocarcinoma. AUTOPSY AND CASE REPORTS 2019; 10:e2019115. [PMID: 32039057 PMCID: PMC6945306 DOI: 10.4322/acr.2019.115] [Citation(s) in RCA: 8] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/22/2018] [Accepted: 02/22/2019] [Indexed: 12/14/2022] Open
Abstract
Tailgut cysts (TGCs) are rare congenital entities arising from remnants of the embryological postanal primitive gut. Malignancy in TGCs is rare, with the majority being adenocarcinomas and carcinoid tumors. A search of the published literature yielded only 27 cases of adenocarcinoma developing in TGCs. We described the case of a 54-year-old female who presented with complaints of pelvic and perineal pain of several weeks. After the initial work-up, a mass in the right presacral location was diagnosed. She underwent radical resection of the tumor, using a posterior approach. The lesion was removed en bloc with the middle rectum, coccyx, and sacrum (S4-S5). The histopathologic examination revealed an adenocarcinoma arising in a TGC, and the patient received adjuvant chemoradiotherapy. Our case underlines that diagnosing a TGC is difficult as it is a rare congenital lesion. Clinical examination may be challenging as TGCs present with various symptoms, which can mimic other commonly proctologic disorders. Patients should be referred to a tertiary center with experience in pelvic surgery and must be managed by a multidisciplinary approach to maximize successful treatment. The recommended treatment is surgical excision given the malignant potential of TGCs and their risk of causing local complications.
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Affiliation(s)
- Pedro Martins
- Instituto Português de Oncologia do Porto Francisco Gentil, Entidade Pública Empresarial, Surgical Oncology Service. Porto, Portugal
| | - Rita Canotilho
- Instituto Português de Oncologia do Porto Francisco Gentil, Entidade Pública Empresarial, Surgical Oncology Service. Porto, Portugal
| | - Mariana Peyroteo
- Instituto Português de Oncologia do Porto Francisco Gentil, Entidade Pública Empresarial, Surgical Oncology Service. Porto, Portugal
| | - Mariana Afonso
- Instituto Português de Oncologia do Porto Francisco Gentil, Entidade Pública Empresarial, Anatomic Pathology Service. Porto, Portugal
| | - Augusto Moreira
- Instituto Português de Oncologia do Porto Francisco Gentil, Entidade Pública Empresarial, Surgical Oncology Service. Porto, Portugal
| | - Abreu de Sousa
- Instituto Português de Oncologia do Porto Francisco Gentil, Entidade Pública Empresarial, Surgical Oncology Service. Porto, Portugal
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4
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Roy SP, Khalessi A, Phan-Thien KC. Robotic resection of a multicystic tailgut cyst. BMJ Case Rep 2019; 12:12/9/e231286. [PMID: 31511270 DOI: 10.1136/bcr-2019-231286] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/17/2022] Open
Abstract
A 29-year-old woman with recurrent pelvic pain that progressed post partum was diagnosed with a multicystic pararectal lesion on ultrasound and CT scan. Physiology was conducted to establish a preoperative function and pudendal nerve integrity. The lesion was resected using a Da Vinci Xi robotic system. She recovered uneventfully with complete resolution of her symptoms. Hindgut cysts most often arise in the presacral space as the result of incomplete embryogenesis. Patients may present with various non-specific symptoms. Although the majority are benign, resection is recommended, as there is a 30%-43% risk of malignancy.
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Affiliation(s)
- Susmit Prosun Roy
- Department of Medicine and Surgery, University of New South Wales - Randwick Campus, Randwick, New South Wales, Australia.,Department of General Surgery, Saint George Hospital, Kogarah, New South Wales, Australia
| | - Amirala Khalessi
- Department of Surgery, Hurstville Private, Hurstville, New South Wales, Australia
| | - Kim-Chi Phan-Thien
- Department of Colorectal Surgery, Saint George Hospital, Kogarah, New South Wales, Australia
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5
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Nicoll K, Bartrop C, Walsh S, Foster R, Duncan G, Payne C, Carden C. Malignant transformation of tailgut cysts is significantly higher than previously reported: systematic review of cases in the literature. Colorectal Dis 2019; 21:869-878. [PMID: 30932326 DOI: 10.1111/codi.14628] [Citation(s) in RCA: 24] [Impact Index Per Article: 4.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/08/2018] [Accepted: 03/16/2019] [Indexed: 02/08/2023]
Abstract
AIM The best treatment for tailgut cysts has not been firmly established. We report a systematic review of the cases in the available literature in order to provide an evidence base for treatment. METHOD A systematic search of articles wholly or partly in English was made of PubMed, Embase and Google Scholar; additional studies were discovered by searching reference lists and contacting authors directly. Search terms 'tailgut cyst', 'tail gut cyst', 'retrorectal hamartoma' and 'retrorectal tumour' were used for case reports or case series; no publication date restrictions were imposed. Only studies with histological confirmation of diagnosis and reporting the age and gender of patients were included. Papers were excluded by consensus between the first two authors. RESULTS A total of 196 individual cases were analysed in detail including 51 cases of neoplasia. The overall rate of neoplastic transformation was 26.6%. Although the male:female cyst incidence ratio was 1:4, men over 18 had a significantly greater relative risk of neoplasm at 1.94 (P = 0.0055). Radiological evidence of nodular thickening of the cyst wall significantly increased the relative risk of the presence of cancer (P = 0.0023). CONCLUSIONS Current orthodoxy that these are not dangerous embryological remnants is unfounded and may be false. The available data suggest the risk of malignant transformation is high and will apply to any residual tissue after excision. The same rationale behind total mesorectal excision in rectal cancer applies to tailgut cysts. Consequently they should be resected with similar oncological margins.
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6
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Aljuhani F, Almunami B, Alsamahi R, Malibary N, Algaithy Z. Alcohol injection for nonsurgical management of tailgut cyst in a middle-aged woman: A case report. Clin Case Rep 2019; 7:1233-1237. [PMID: 31183101 PMCID: PMC6552934 DOI: 10.1002/ccr3.2205] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/19/2018] [Revised: 03/15/2019] [Accepted: 03/27/2019] [Indexed: 12/20/2022] Open
Abstract
Managing recurrent tailgut cyst in a patient who is refraining from the definitive surgical en bloc resection can be challenging. Therefore, in this case report we outlined a less invasive approach which is computed tomography-guided aspiration with alcohol injection which resulted in prolongation of symptoms free period in our patient.
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Affiliation(s)
- Farrah Aljuhani
- Faculty of MedicineKing Abdulaziz UniversityJeddahSaudi Arabia
| | | | - Rawan Alsamahi
- Faculty of MedicineKing Abdulaziz UniversityJeddahSaudi Arabia
| | - Nadim Malibary
- Faculty of MedicineKing Abdulaziz UniversityJeddahSaudi Arabia
| | - Zuhoor Algaithy
- Faculty of MedicineKing Abdulaziz UniversityJeddahSaudi Arabia
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7
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Demirel AH, Cetin E, Temiz A. Squamous cell carcinoma arising in a sacrococcygeal tailgut cyst. An Bras Dermatol 2018; 93:733-735. [PMID: 30156628 PMCID: PMC6106680 DOI: 10.1590/abd1806-4841.20187618] [Citation(s) in RCA: 9] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/05/2017] [Accepted: 10/13/2017] [Indexed: 02/07/2023] Open
Abstract
We report a case of squamous cell carcinoma originated from a sacrococcygeal tailgut cyst in a 73-year-old female patient. Tailgut cysts are generally multilocal and have a layer of either columnar, squamous or transitional epithelium, or a combination of these. This case was treated with surgical excision and radiotherapy. Cancer presentation of a congenital abnormality in old age is a rare entity. This report is the first case of squamous cell carcinoma developing in a tailgut cyst without any synchronization, as an isolated (pure) pathology.
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Affiliation(s)
- Arif Hakan Demirel
- Division of Surgery, Yenimahalle Education and Research Hospital,
Faculty of Medicine, Yıldırım Beyazıt University,
Ankara, Turkey
| | - Eren Cetin
- Department Radiation Oncology, Faculty of Medicine, Gazi University,
Ankara, Turkey
| | - Atiye Temiz
- Division of Pathology, Yenimahalle Education and Research Hospital,
Faculty of Medicine, Yıldırım Beyazıt University,
Ankara, Turkey
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8
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Diagnosis of Tailgut Cyst in Gynecologic Patients: Systematic Review of the Literature. JOURNAL OF ENDOMETRIOSIS AND PELVIC PAIN DISORDERS 2017. [DOI: 10.5301/jeppd.5000293] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/20/2022]
Abstract
Tailgut cysts, also called retrorectal cystic hamartomas, are rare findings that usually occur in the retrorectal space. The incidence is estimated to be about 1 in 40,000.The aim of our review is to evaluate the potential pitfalls in the diagnosis of this rare pathologic condition, according to diagnostic procedures for this rare but misleading and subtle entity.We conducted a Medline literature review of the English literature discussing tailgut cysts in female patients. We concentrated on imaging technology used for diagnosis in gynecologic patients. For our search, we used the key words “tailgut cyst”, “retrorectal cystic hamartoma”, “gynecology”, focusing on clinical presentation, imaging technology used for diagnosis, presence of malignancy, and misdiagnosis with more common gynecologic pathology.Our search revealed 143 articles during the years 1975–2016 and, of these, 30 articles describing 91 female patients were considered.We concluded that tailgut cyst should be included in the differential diagnosis of pelvic lesions in gynecologic patients.
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9
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Morotti A, Busso M, Consiglio Barozzino M, Cinardo P, Angelino V, Familiari U, Veltri A, Guerrasio A. Detection and management of retroperitoneal cystic lesions: A case report and review of the literature. Oncol Lett 2017; 14:1602-1608. [PMID: 28789385 DOI: 10.3892/ol.2017.6323] [Citation(s) in RCA: 16] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/12/2016] [Accepted: 11/04/2016] [Indexed: 12/25/2022] Open
Abstract
The identification of cystic lesions within the retroperitoneal space is a rare event that poses clinicians the challenge of a difficult diagnosis and disease management. Retroperitoneal cystic lesions account for a group of lesions that range from common benign lesions (e.g., lymphoceles developing as a surgical complication) to rare aggressive malignant neoplasms. Currently, in the majority of cases, image-guided procedures allow for a pathological diagnosis to be achieved in these challenging lesions, thus offering the chance of an appropriate treatment; however, the overall clinical assessment of retroperitoneal cysts is highly demanding. The present study reports the management of a representative clinical case, presenting with a voluminous cystic mass able to dislocate cave vein, whose diagnosis was preceded by a deep vein thrombosis. Computed tomography-scan and ultrasound guided percutaneous drainage were performed to achieve the diagnosis. Following the discussion of the current case report, a review of the pathological and radiological characteristics of retroperitoneal cystic lesions is presented.
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Affiliation(s)
- Alessandro Morotti
- Division of Internal Medicine, University of Turin, Orbassano, I-10043 Turin, Italy.,Department of Clinical and Biological Sciences, University of Turin, Orbassano, I-10043 Turin, Italy
| | - Marco Busso
- Department of Oncology, University of Turin and San Luigi Hospital, Orbassano, I-10043 Turin, Italy
| | - Maria Consiglio Barozzino
- Department of Clinical and Biological Sciences, University of Turin, Orbassano, I-10043 Turin, Italy
| | - Paola Cinardo
- Department of Clinical and Biological Sciences, University of Turin, Orbassano, I-10043 Turin, Italy
| | - Valeria Angelino
- Department of Oncology, University of Turin and San Luigi Hospital, Orbassano, I-10043 Turin, Italy
| | - Ubaldo Familiari
- Department of Pathology, San Luigi Hospital, Orbassano, I-10043 Turin, Italy
| | - Andrea Veltri
- Department of Oncology, University of Turin and San Luigi Hospital, Orbassano, I-10043 Turin, Italy
| | - Angelo Guerrasio
- Division of Internal Medicine, University of Turin, Orbassano, I-10043 Turin, Italy.,Department of Clinical and Biological Sciences, University of Turin, Orbassano, I-10043 Turin, Italy
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10
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Seydafkan S, Shibata D, Sanchez J, Tran ND, Leon M, Coppola D. Pathology Report: Presacral Noncommunicating Enteric Duplication Cyst. Cancer Control 2017; 23:170-4. [PMID: 27218796 DOI: 10.1177/107327481602300213] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/13/2022] Open
Abstract
BACKGROUND Gastrointestinal (GI) tract duplication cysts or enteric duplication cysts are rare congenital malformations sometimes found on the mesenteric aspect of segments of the alimentary tract. Enteric duplication cysts are lined by normal GI epithelium and may be classified as foregut, mid-gut, and hindgut cysts. Except in very rare cases of retroperitoneal enteric duplication cysts, these cysts communicate with the GI tract and share a common blood supply. Concurrent congenital malformations are not uncommon and malignant transformation within enteric duplication cysts has also been reported. METHODS We describe a case of a noncommunicating enteric duplication cyst in a 52-year-old woman. RESULTS The patient presented with a presacral cystic mass requiring frequent drainage procedures that was primarily believed to be of neural origin. Upon resection, the lesion contained heterotopic tissue, including ciliated bronchial epithelium, squamous and transitional epithelia, and pancreatic and gastric tissue. Focal, low-grade intestinal adenoma was present, but malignancy was not detected in this case. CONCLUSION To our knowledge, this is the sixth reported case of a noncommunicating enteric duplication cyst in the English medical literature.
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Affiliation(s)
- Shabnam Seydafkan
- Department of Anatomic Pathology, Moffitt Cancer Center, Tampa, FL 33612, USA.
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11
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Haydar M, Griepentrog K. Tailgut cyst: A case report and literature review. Int J Surg Case Rep 2015; 10:166-8. [PMID: 25853843 PMCID: PMC4430219 DOI: 10.1016/j.ijscr.2015.03.031] [Citation(s) in RCA: 19] [Impact Index Per Article: 1.9] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/31/2014] [Revised: 03/11/2015] [Accepted: 03/12/2015] [Indexed: 12/19/2022] Open
Abstract
Incidence, embryological origin and formation of a tailgut cyst. Clinical presentation, radiological findings, and diagnosis of a tailgut cyst. Description of our patient’s medical course and surgical treatment. Surgical indications, pros and cons of different surgical approaches. Introduction Tailgut cysts are rare diseases but can be challenging to manage. Presentation of case This is a case report of a tailgut cyst, in a 42 year old female. The cyst was incidentally detected on imaging, and treated with Kraske surgical approach. Discussion Hereby, we review the relevant information present in the medical literature about the incidence, methods of diagnosis, treatment options and management plans. Conclusion Surgical management should be tailored individually for each patient, based on the advantages and disadvantages of each surgical approach.
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Affiliation(s)
- Mazen Haydar
- Department of Visceral and Vascular Surgery, Kreiskrankenhaus Stollberg gGmbH, Jahnsdorfer Straße 7, Stollberg 09366, Germany.
| | - Kai Griepentrog
- Department of Visceral and Vascular Surgery, Kreiskrankenhaus Stollberg gGmbH, Jahnsdorfer Straße 7, Stollberg 09366, Germany.
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12
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13
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Misawa SI, Horie H, Yamaguchi T, Kobayashi S, Kumano H, Lefor AT, Yasuda Y. A Unique Retrorectal Tumor With Neuroendocrine Differentiation. Int J Surg Pathol 2013; 21:271-7. [DOI: 10.1177/1066896913476738] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/16/2023]
Abstract
Retrorectal or presacral tumors are very rare. We report a unique case of a retrorectal tumor with neuroendocrine differentiation, consisting of high- and low-grade components. A 53-year-old woman treated for a perianal abscess at another clinic was referred to our hospital for continued anal pain. Digital rectal examination identified a soft tumor with a smooth surface in the lower rectum. Pelvic computed tomography and magnetic resonance imaging detected a large cystic tumor measuring 8 cm in diameter in the retrorectal space of the pelvis. The border between the tumor and rectal wall, levator ani and vaginal wall was obscure. Fine-needle aspiration cytology was highly suggestive of carcinoma. Abdominoperineal resection was performed, and the tumor was histologically diagnosed as a neuroendocrine carcinoma based on immunohistochemical staining. No previous case has been reported with cystic growth of a neuroendocrine carcinoma in the retrorectal space.
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Affiliation(s)
| | | | | | | | | | - Alan T. Lefor
- Jichi Medical University, Shimotsuke, Tochigi, Japan
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14
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Chhabra S, Wise S, Maloney-Patel N, Rezac C, Poplin E. Adenocarcinoma associated with tail gut cyst. J Gastrointest Oncol 2013; 4:97-100. [PMID: 23450681 DOI: 10.3978/j.issn.2078-6891.2012.043] [Citation(s) in RCA: 11] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Key Words] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/20/2012] [Accepted: 08/26/2012] [Indexed: 12/14/2022] Open
Abstract
Primary adenocarcinomas of the presacral (retrorectal) space are rare. The diagnosis is usually delayed because of non-specific symptoms, and is made after a biopsy or surgery. These carcinomas arise from cystic lesions developing from remnants of the embryological postanal gut containing mucous-secreting epithelium, known as tail gut cysts. The potential for infection, perianal fistulas and most importantly, malignant change warrants an early complete surgical resection. From an oncologist's perspective, the management of these carcinomas has varied, and has included adjuvant chemotherapy and/or radiation therapy. We describe here a rare case of adenocarcinoma associated with a tail gut cyst that was discovered incidentally and resected by a posterior approach (Kraske procedure). The patient has had clinical and periodic radiologic surveillance without any evidence of cancer recurrence for over a year and a half.
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15
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Delemer M, Detchev R, Dugue T, Gosset P, Houzé de l’Aulnoit D. L’hamartome kystique rétrorectal (HKRR) : à propos d’un cas réséqué par abord vaginal et revue de la littérature. ACTA ACUST UNITED AC 2011; 40:569-71. [DOI: 10.1016/j.jgyn.2010.12.009] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/11/2010] [Revised: 12/09/2010] [Accepted: 12/23/2010] [Indexed: 12/28/2022]
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16
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McCawley N, Hanly A, Solon G, Shilling C, Deasy J, Kay EW, McNamara DA. Adenocarcinoma arising in a retrorectal teratoma: case report and review of the literature. Colorectal Dis 2011; 13:e312-3. [PMID: 20958914 DOI: 10.1111/j.1463-1318.2010.02461.x] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 01/17/2023]
Affiliation(s)
- N McCawley
- Department of Colorectal Surgery, Beaumont Hospital, Dublin, Ireland
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17
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Abstract
Purpose. This review summarizes the more prevalent soft tissue
tumours arising in the retroperitoneum and highlights some recent fundamental and
diagnostic developments relevant to mesenchymal tumours. Discussion. The retroperitoneum is an underestimated site for benign
and malignant neoplastic disease, and represents the second most common site of origin
of primary malignant soft tissue tumours (sarcomas) after the deep tissues of the lower
extremity. In contrast to the predominance of benign soft tissue lesions over malignant
sarcomas elsewhere, retroperitoneal mesenchymal lesions are far more likely to be malignant.
The differential diagnosis is primarily with the more common lymphoproliferative and
parenchymatous epithelial lesions arising in this area, and with metastatic disease from known
or unknown primary sites elsewhere.The most prevalent mesenchymal tumours at this site are
of a lipomatous, myogenic or neural nature.Their generally late clinical presentation and poorly
accessible location provides numerous clinical challenges; optimal radiological imaging and
a properly performed biopsy are essential cogs in the management route. Histopathological
diagnosis may be complicated, but has been aided by developments in the fields of
immunohistochemistry and tumour (cyto)genetics. Despite significant advances in oncological
management protocols, the prognosis remains generally less favourable than for similar
tumours at more accessible sites.
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18
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Gaud U, Goyal T, Shukla M, Kumar V, Pandey M. Synchronous occurrence of adenocarcinoma of the rectum with squamous cell carcinoma of a retrorectal cyst: report of a case and review of the literature. BMJ Case Rep 2009; 2009:bcr12.2008.1398. [PMID: 21686338 DOI: 10.1136/bcr.12.2008.1398] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/23/2022] Open
Abstract
Retrorectal cysts are rare congenital anomalies which are more common in females. Rarely, malignant transformation can occur in these cysts. We report here the case of a middle-aged man who had synchronous presentation of adenocarcinoma of the rectum and squamous cell carcinoma in a retrorectal cyst. The patient presented with rectal bleeding and constipation, and underwent abdominoperineal resection and complete excision of the cyst. Postoperative histopathological examination showed adenocarcinoma of the rectum and squamous cell carcinoma of the cyst. The patient received adjuvant chemotherapy and after 18 months of follow-up is free of any local or metastatic disease. Synchronous occurrence of rectal cancer with carcinoma in a retrorectal cyst has not been previously reported in the literature in English.
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Affiliation(s)
- Umakant Gaud
- Institute of Medical Sciences, Surgical Oncology, Banaras Hindu University, Varanasi, UP, 221005, India
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19
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Vestigial retrorectal cystic tumors in adults: A review of 30 cases. ACTA ACUST UNITED AC 2008; 32:769-78. [DOI: 10.1016/j.gcb.2008.03.011] [Citation(s) in RCA: 25] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/14/2007] [Revised: 02/28/2008] [Accepted: 03/01/2008] [Indexed: 12/24/2022]
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Tailgut cysts: MRI findings. Eur Radiol 2008; 18:2586-93. [PMID: 18566821 DOI: 10.1007/s00330-008-1028-4] [Citation(s) in RCA: 36] [Impact Index Per Article: 2.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/30/2007] [Revised: 03/18/2008] [Accepted: 04/09/2008] [Indexed: 01/16/2023]
Abstract
Magnetic resonance imaging (MRI) features of 11 surgically resected pelvic tailgut cysts were analyzed with reference to histopathologic and clinical data. Homogeneity, size, location, signal intensity, appearance and presence of septa and/or nodules and/or peripheral rim and involvement of surrounding structures were studied. Histological examination demonstrated 11 tailgut cysts (TGC), including one infected TGC and one TGC with a component of adenocarcinoma. Lesions (3-8 cm in diameter) were exclusively or partly retrorectal in all cases but one, with an extension down the anal canal in five cases. Lesions were multicystic in all patients but one. On T1-weighted MR images, all cystic lesions contained at least one hyperintense cyst. The peripheral rim of the cystic lesion was regular and non or moderately enhancing in all cases but the two complicated TGC. Nodular peripheral rim and irregular septa were seen in the degenerated TGC. Marked enhancement of the peripheral structures was noted in the two complicated TGC. Pelvic MRI is a valuable tool in the preoperative evaluation of TGC.
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21
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Vega Menéndez D, Quintáns Rodríguez A, Hernández Granados P, Nevado Santos M, García Sabrido JL, Rueda Orgaz JA, Ochando F, Ojeda Pérez F, Loinaz C. Hamartomas quísticos retrorrectales. Cir Esp 2008; 83:53-60. [DOI: 10.1016/s0009-739x(08)70506-2] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/29/2022]
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22
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Garcia-Donas J, Rodriguez N, Jara C, Urioste M, Nevado M, Cañamero M, Cuartero V, Albillos J, Vega D, Quintans A. Retrorectal cystic hamartoma as benign cause of CA 19-9 elevation. J Clin Oncol 2007; 25:4012-4. [PMID: 17761987 DOI: 10.1200/jco.2007.12.4214] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/17/2023] Open
Affiliation(s)
- Jesus Garcia-Donas
- Department of Medical Oncology, Fundación Hospital Alcorcón, Alcorcón, Spain
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23
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Rees CM, Woodward M, Grier D, Cusick E. Rectal duplication cyst: a combined abdominal and endoanal operative approach. Pediatr Surg Int 2007; 23:369-71. [PMID: 17013624 DOI: 10.1007/s00383-006-1807-4] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Accepted: 09/11/2006] [Indexed: 01/17/2023]
Abstract
Rectal duplication cysts are rare, comprising <5% of all gastrointestinal duplications. Early excision is the treatment of choice and a number of surgical approaches have been described. We present a 3-week-old infant with a 3 cm cyst that was excised using a previously unreported combined abdominal and endoanal approach.
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Affiliation(s)
- Clare M Rees
- Department of Paediatric Surgery, Bristol Royal Hospital for Children, Bristol, UK
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24
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Piura B, Rabinovich A, Sinelnikov I, Delgado B. Tailgut cyst initially misdiagnosed as ovarian tumor. Arch Gynecol Obstet 2005; 272:301-3. [PMID: 16041543 DOI: 10.1007/s00404-005-0012-3] [Citation(s) in RCA: 13] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/25/2005] [Accepted: 01/31/2005] [Indexed: 01/17/2023]
Abstract
INTRODUCTION Tailgut cyst (retrorectal cystic hamartoma) is an uncommon congenital lesion that arises from remnants of the embryonic post-anal gut. It is usually benign and located in the retrorectal/presacral space. CASE REPORT A 45-year-old woman presented with lower abdominal discomfort, urinary frequency and right-sided pelvic mass. Digital rectal examination and computerized tomography scan demonstrated a multicystic complex mass occupying the region of the right ovary and extending into the retrorectal/presacral space. The initial diagnosis was neoplasm of the right ovary. At laparotomy, the mass was lying free from apparently normal internal genital organs. Excision of the mass from the right pelvis and retrorectal/presacral space and total abdominal hysterectomy were performed. Microscopic examination revealed that the wall of the cystic mass consists of a lining epithelium composed of columnar and squamous epithelium and a stroma composed of fibrous tissue containing scattered discontinuous bundles of smooth muscle fibers. Based on these histological findings, the final diagnosis was tailgut cyst. CONCLUSION Tailgut cyst is an uncommon entity that should be included in the differential diagnosis of retrorectal/presacral mass. The treatment of choice is complete surgical excision.
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Affiliation(s)
- Benjamin Piura
- Unit of Gynecologic Oncology, Department of Obstetrics and Gynecology, Soroka Medical Center, Ben-Gurion University of the Negev, 151, Beer-Sheva, 84101, Israel.
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25
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Cho BC, Kim NK, Lim BJ, Kang SO, Sohn JH, Roh JK, Choi ST, Kim SA, Park SE. A carcinoembryonic antigen-secreting adenocarcinoma arising in tailgut cyst: clinical implications of carcinoembryonic antigen. Yonsei Med J 2005; 46:555-61. [PMID: 16127782 PMCID: PMC2815842 DOI: 10.3349/ymj.2005.46.4.555] [Citation(s) in RCA: 20] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 01/26/2023] Open
Abstract
Tailgut cysts (TGCs) are rare congenital cysts that occur in the retrorectal or presacral spaces. Although most tailgut cysts have been reported as benign, there have been at least 9 cases associated with malignant change. We report herein on an unusual case of a 40-year-old woman with a carcinoembryonic antigen (CEA)-producing adenocarcinoma arising within a TGC who underwent surgical resection and local radiation therapy. Despite the complete resection, metastatic adenocarcinoma developed five months after surgery. CEA-producing adenocarcinoma from a TGC is extremely rare and only two cases, including this case, have been reported in the English medical literature. Besides CEA, the serum levels of CA 19-9 became markedly elevated in this patient. Given that the serum CEA level decreased to the normal range after complete resection of tumor and that the tumor recurrence was associated with a rebound of the CEA serum level, our case shows that serial measurements of serum CEA can be used for treatment planning and for assessing the patient's treatment response for this rare disease.
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Affiliation(s)
- Byoung Chul Cho
- Department of Internal Medicine, Yonsei University College of Medicine, Seoul, Korea
| | - Nam Kyu Kim
- Department of Surgery, Yonsei University College of Medicine, Seoul, Korea
| | - Beom Jin Lim
- Department of Pathology, Yonsei University College of Medicine, Seoul, Korea
| | - Sang Ook Kang
- Department of Surgery, Yonsei University College of Medicine, Seoul, Korea
| | - Ju Hyuk Sohn
- Department of Internal Medicine, Yonsei University College of Medicine, Seoul, Korea
| | - Jae Kyung Roh
- Department of Internal Medicine, Yonsei University College of Medicine, Seoul, Korea
| | - Sang Tae Choi
- Department of Internal Medicine, Yonsei University College of Medicine, Seoul, Korea
| | - Sung Ai Kim
- Department of Internal Medicine, Yonsei University College of Medicine, Seoul, Korea
| | - Se Eun Park
- Department of Internal Medicine, Yonsei University College of Medicine, Seoul, Korea
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26
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Killingsworth C, Gadacz TR. Tailgut Cyst (Retrorectal Cystic Hamartoma): Report of a Case and Review of the Literature. Am Surg 2005. [DOI: 10.1177/000313480507100810] [Citation(s) in RCA: 44] [Impact Index Per Article: 2.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/16/2023]
Abstract
Tailgut cysts are rare congenital lesions arising from remnants of normally regressing postanal primitive gut. They often present in middle-aged women with perirectal symptoms and a retrorectal multicystic mass. These cysts have occasionally shown malignant transformation. We report a case of a tailgut cyst occurring in a 25-year-old African-American female. The differential diagnosis of a retrorectal mass is briefly explored, and the etiology, diagnostic strategy, and surgical approach for tailgut cysts is examined. We also report an extensive literature review to examine clinical characteristics and surgical data for 43 cases of tailgut cysts spanning 16 years.
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Affiliation(s)
| | - Thomas R. Gadacz
- Department of Surgery, Medical College of Georgia, Augusta, Georgia
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27
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Andea AA, Klimstra DS. Adenocarcinoma arising in a tailgut cyst with prominent meningothelial proliferation and thyroid tissue: case report and review of the literature. Virchows Arch 2005; 446:316-21. [PMID: 15731926 DOI: 10.1007/s00428-004-1178-y] [Citation(s) in RCA: 23] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/20/2004] [Accepted: 11/10/2004] [Indexed: 01/26/2023]
Abstract
Tailgut cysts (retrorectal cystic hamartomas) are rare lesions occurring in the retrorectal/presacral space of young women, considered to be of developmental origin. In this study, we report the clinicopathological characteristics of an adenocarcinoma developing in an unusual tailgut cyst. A 47-year-old female presented with a 3-month history of an enlarging presacral mass, extending into the gluteal area. The patient underwent surgical resection. Microscopic examination revealed a mucinous adenocarcinoma with intestinal features arising in a tailgut cyst. In addition to the usual components previously described in this lesion (cuboidal, columnar, ciliated or squamous epithelial lining with scattered smooth muscle fibers in the wall), our case was unique in that it was associated with a meningothelial proliferation and benign thyroid tissue with oncocytic changes. These additional features bring into question the proposed hindgut origin of tailgut cysts.
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Affiliation(s)
- Aleodor A Andea
- Memorial Sloan-Kettering Cancer Center, Department of Pathology, 1275 York Avenue, New York, NY 10021, USA
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28
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Shivnani AT, Small W, Benson A, Rao S, Talamonti MS. Adenocarcinoma Arising in Rectal Duplication Cyst: Case Report and Review of the Literature. Am Surg 2004. [DOI: 10.1177/000313480407001116] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/30/2022]
Abstract
Duplication cyst of the gastrointestinal (GI) tract is a rare congenital anomaly, and rectal duplication cysts comprise a small fraction these cases. Most patients present for the first time in adulthood, and the origin of rectal duplication cysts is unclear. Prior series document malignant transformation in approximately 20 per cent of cases. The following case report describes a carcinoma arising in a rectal duplication cyst. Given the lack of data demonstrating adequate control for patients with adenocarcinoma arising in a rectal duplication cyst and our experience with this patient, we recommend all patients undergo multidisciplinary evaluation prior to any therapy.
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Affiliation(s)
- Anand T. Shivnani
- Division of Radiation Oncology, Robert H. Lurie Comprehensive Cancer Center, Northwestern University, Chicago, Illinois
| | - William Small
- Division of Radiation Oncology, Robert H. Lurie Comprehensive Cancer Center, Northwestern University, Chicago, Illinois
| | - Al Benson
- Division of Hematology/Oncology, Robert H. Lurie Comprehensive Cancer Center, Northwestern University, Chicago, Illinois
| | - Sambasiva Rao
- Department of Pathology, Robert H. Lurie Comprehensive Cancer Center, Northwestern University, Chicago, Illinois
| | - Mark S. Talamonti
- Division of Hematology/Oncology, Robert H. Lurie Comprehensive Cancer Center, Northwestern University, Chicago, Illinois
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29
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Song DE, Park JK, Hur B, Ro JY. Carcinoid tumor arising in a tailgut cyst of the anorectal junction with distant metastasis: a case report and review of the literature. Arch Pathol Lab Med 2004; 128:578-80. [PMID: 15086297 DOI: 10.5858/2004-128-578-ctaiat] [Citation(s) in RCA: 44] [Impact Index Per Article: 2.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/06/2022]
Abstract
Tailgut cyst is a rare congenital presacral lesion and is believed to arise from the persistent remnants of the postanal gut. Malignancy occurring in a tailgut cyst is extremely rare, and to our knowledge only 5 cases of carcinoid tumor arising in a tailgut cyst have been reported in the literature to date. We report a sixth case of carcinoid tumor arising in a tailgut cyst. The patient was a 41-year-old woman who presented with perianal pain. Sigmoidoscopy showed a 2-cm submucosal mass located 4 cm above the anal verge. The mass was a multilocular cyst with gray-tan solid portions. The cyst was lined by ciliated columnar, squamous, and transitional epithelia with small foci of carcinoid tumor in the cystic wall. The carcinoid tumor showed a trabecular growth pattern with uniform oval or round cells containing fine chromatin and positive immunoreactivity for chromogranin, synaptophysin, and cytokeratin. This case was unique because the tumor occurred at the anorectal junction, not in the retrorectal space, and unlike previously reported cases showed aggressive behavior and distant metastases.
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Affiliation(s)
- Dong Eun Song
- Department of Pathology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
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30
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Abstract
Retrorectal lesions are rare entities. We report the complete clinicopathological details of an unusual retrorectal lesion composed of low-grade endometrioid adenocarcinoma and propose that this is a malignant change in pluripotent cells arising within a long-standing retrorectal tailgut lesion. To the best of our knowledge, this is the first case report of this malignancy in a retrorectal tailgut cyst.
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Affiliation(s)
- Selliah C Kanthan
- Department of Pathology, College of Medicine, University of Saskatchewan, Room 2868, G Wing, Royal University Hospital, Saskatoon, Saskatchewan S7N 0W8, Canada.
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31
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Menassa-Moussa L, Kanso H, Checrallah A, Abboud J, Ghossain M. CT and MR findings of a retrorectal cystic hamartoma confused with an adnexal mass on ultrasound. Eur Radiol 2004; 15:263-6. [PMID: 15662478 DOI: 10.1007/s00330-004-2330-4] [Citation(s) in RCA: 17] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/16/2003] [Revised: 03/08/2004] [Accepted: 04/01/2004] [Indexed: 01/16/2023]
Abstract
We describe the imaging features of a tailgut cyst mistaken for an adnexal mass. A pelvic ultrasound in a 28-year-old woman showed a 10-cm hypoechoic left pelvic mass. Having not seen the left ovary, the radiologist concluded that the mass was an endometrioma. CT disclosed a retrorectal cystic lesion with wall calcifications and internal septa. MR confirmed the extra-ovarian location of the tumor, which was hyperintense on T2-weighted images and had an intermediate signal on T1-weighted images. Surgery revealed a retrorectal cystic hamartoma. Radiological diagnosis of a tailgut cyst requires first correct localization of the tumor and then differentiation from other retrorectal masses.
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Affiliation(s)
- L Menassa-Moussa
- Department of Radiology, Hôtel Dieu de France, Boulevard Alfred Naccache, Achrafieh, Beirut, Lebanon
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32
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33
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Abstract
Tailgut cysts are rare congenital multicystic lesions preferentially occurring in the retrorectal space. They are assumed to arise from the remnant of embryonic tailgut and occasionally exhibit malignant transformation. The authors herein describe the clinical, radiologic, and pathologic features of an unusual perirenal tailgut cyst with an emphasis on its histogenesis and distinction from other intraabdominal cystic lesions.
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Affiliation(s)
- Ming-Tse Sung
- Department of Pathology, Chang Gung Memory Hospital, Kaohsiung Medical Center, Chang Gung University, Taiwan, China
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34
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Murao K, Fukui Y, Numoto S, Urano Y. Tailgut cyst as a subcutaneous tumor at the coccygeal region. Am J Dermatopathol 2003; 25:275-7. [PMID: 12775995 DOI: 10.1097/00000372-200306000-00018] [Citation(s) in RCA: 10] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/17/2023]
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35
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Satyadas T, Davies M, Nasir N, Halligan S, Akle CA. Tailgut cyst associated with a pilonidal sinus: an unusual case and a review. Colorectal Dis 2002; 4:201-204. [PMID: 12780617 DOI: 10.1046/j.1463-1318.2002.00345.x] [Citation(s) in RCA: 12] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/08/2023]
Abstract
Tailgut cysts are rare congenital abnormalities in the retrocaecal/presacral region. Clinical diagnosis is difficult and delayed and they can present in childhood and adult life with a variety of clinical symptoms and complications. Differential diagnoses include, rectal duplication cysts, cystic teratoma, epidermal cyst, anal gland cyst and anal gland carcinoma. Magnetic resonance imaging has recently become the modality of choice to image these cysts. Although Tailgut cysts rarely undergo malignant transformation, early surgical resection is presently considered the treatment of choice. Here we report the case of a 34 year old gentleman with a Tailgut cyst associated with a Pilonidal sinus, and review of the literature. We believe that the high incidence of complications associated with operations in the presacral region should be weighed against the generally benign course of these lesions, especially with the quality of modern imaging technology.
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Affiliation(s)
- T Satyadas
- Royal Free Hospital and University Medical School, London, UK, The London Clinic, London, UK, St Mark's Hospital, London, UK
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36
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Moreira AL, Scholes JV, Boppana S, Melamed J. p53 Mutation in adenocarcinoma arising in retrorectal cyst hamartoma (tailgut cyst): report of 2 cases--an immunohistochemistry/immunoperoxidase study. Arch Pathol Lab Med 2001; 125:1361-4. [PMID: 11570917 DOI: 10.5858/2001-125-1361-pmiaai] [Citation(s) in RCA: 26] [Impact Index Per Article: 1.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/14/2022]
Abstract
Retrorectal cyst hamartoma (RCH) is a rare benign cystic lesion located in the retrorectal space. Malignancy arising in such lesions is very uncommon. In this study, 2 cases of mucinous adenocarcinoma arising in RCH are presented. In one case, dysplastic epithelium lined the cyst wall, surrounding the area of carcinoma and suggesting a dysplasia-carcinoma progression in RCH. Adenocarcinoma and the dysplastic epithelium were strongly positive for p53 and Ki-67 and showed negative staining for p21 by immunohistochemistry. These findings are suggestive of a mutation in the p53 gene in the adenocarcinoma and in dysplastic epithelium lining the cysts, similar to the dysplasia-carcinoma sequence described for the development of colonic adenocarcinoma.
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Affiliation(s)
- A L Moreira
- Department of Pathology, New York University Medical Center, New York, NY 10016, USA
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37
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Umar T, Mikel JJ, Poller DN. Carcinoma arising in a tailgut cyst diagnosed on fine needle aspiration (FNA) cytology. Cytopathology 2000; 11:129-32. [PMID: 10772013 DOI: 10.1046/j.1365-2303.2000.00215.x] [Citation(s) in RCA: 10] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/17/2023]
Affiliation(s)
- T Umar
- Department of Pathology, Queen Alexandra Hospital, Cosham, Portsmouth, UK
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