Tailgut Cyst in a Child: A Case Report and Review of Literature.

ABSTRACT: Tailgut cysts are rare congenital abnormalities that develop due to failure of embryologic tailgut to involute. This generally manifests as a presacral, retrorectal and multicystic mass. It has a high propensity in female patients including paediatric age group. The clinical presentation varies depending on the age of patient, location and size of the cyst. The symptoms are mostly due to mass effects and rarely complications such as malignancy. Being a rare entity, it is generally misdiagnosed. Differential diagnosis mostly includes rectal duplication cyst, sacrococcygeal teratoma and anterior meningocele. Radiological imaging such as computed tomography scan and magnetic resonance imaging is useful in the diagnosis. The treatment of choice is complete excision to prevent recurrence, infection and malignant degeneration. We have reviewed previous literature and given our valuable information regarding the same.

Management of a complicated colonexposed sacrococcygeal wound after pelvic exenteration: a case report.

DECLARATION OF INTEREST: This work was supported by the Naval Medical University and the University of Shanghai for Science and Technology Joint Projects (2020-RZ04), the Innovative Clinical Research Program of Shanghai Changzheng Hospital (2020YLCYJ-Y16), and the academic project of Naval Medical University (2022QN073). The authors have no conflicts of interest to declare.

Presacral Tailgut Cyst.

A tailgut cyst is a rare benign polycystic congenital lesion in presacral or retrorectal space, when there is failure of involution of tailgut. Clinical presentation may be with or without symptoms of different types related to obstruction, infection, or rarely malignancy. Symptoms may be misleading and atypical, so understanding of characteristics of tailgut cysts is important for precise and early diagnosis to be made for proper treatment and to avoid complications and malignant transformation. Magnetic resonance imaging pelvis was used as diagnostic imaging investigation, but the final confirmation was only done by histopathology. Definitive treatment is surgery, though various surgical approaches are described, surgery is tailored which will suit the individual patient's anatomy and suspected diagnosis of mass. This case review used PubMed and Web of Science databases to search for the studies. We found around 176 articles and selected 77 articles in our survey, with 8 reviews, 31 case reports, and 31 case reports and reviews.

RésuméUn kyste intestinal est une lésion congénitale polykystique bénigne rare dans l'espace présacré ou rétrorectal, en cas d'échec de l'involution de l'intestin grêle. La présentation clinique peut être accompagnée ou non de symptômes de différents types liés à une obstruction, une infection ou, plus rarement, une tumeur maligne. Les symptômes peuvent être trompeurs et atypiques, c'est pourquoi il est important de comprendre les caractéristiques des kystes de l'intestin grêle pour établir un diagnostic précis et précoce afin d'établir un traitement approprié et d'éviter les complications et la transformation maligne. L'imagerie par résonance magnétique du bassin a été utilisée comme examen d'imagerie diagnostique, mais la confirmation finale n'a été faite que par histopathologie. Le traitement définitif est la chirurgie, bien que diverses approches chirurgicales soient décrites, la chirurgie est adaptée à l'anatomie de chaque patient et au diagnostic suspecté de masse. Cette revue de cas a utilisé les bases de données PubMed et Web of Science pour rechercher les études. Nous avons trouvé environ 176 articles et sélectionné 77 articles dans notre enquête, avec 8 revues, 31 rapports de cas et 31 rapports de cas et critiques.

Prenatal diagnosis in estimating the prognosis of sacrococcygeal teratoma.

: Sacrococcygeal teratoma is a rare congenital malformation, the prognosis depends on factors affecting foetal development. The diagnosis is based on ultrasound examination, especially the evaluation of the detailed morphology of the foetus in the 20th week of pregnancy. Therefore, it is crucial to keep looking for ultrasound markers that would prenatally determine the most accurate prognosis for the foetus. Now, we rely on a small number of studies with a predominance of case reports. We offer a literature review of the essential information concerning sacrococcygeal teratoma diagnostics, therapy, and complications of sacrococcygeal teratomas in connection with prenatal diagnosis. It turns out that in cases with a favourable prognosis according to prenatal ultrasound examination and adequate surgical treatment after childbirth, the prognosis of this congenital malformation is excellent.

Endoscopic pilonidal sinus treatment (EPSiT) for sacrococcygeal pilonidal sinus disease: A prospective long-term study.

Objectives: To assess short-term and long-term outcomes of endoscopic pilonidal sinus treatment for pilonidal sinus disease. METHODS: The prospective study was conducted at Shifa International Hospital, Islamabad, Pakistan, from July 2015 to July 2021, and comprised all pilonidal sinus cases undergoing minimal invasive endoscopic pilonidal sinus treatment who were treated by a single surgical team. The primary outcomes were duration of healing, post-operative morbidities, persistence of discharge and recurrence at 1-7 years. The secondary outcomes were operative time, return to work, cosmetic results and patient satisfaction. The patients were observed for wound healing and discharge on follow-up in the out-patient department at 1, 3, 6 and 24 weeks. They were further followed up every year through telephonic survey for persistence or recurrence of symptoms. Patient satisfaction was assessed using the 36-item Short Form Survey questionnaire filled at admission and then at 6 weeks post-surgery. Data was analysed using SPSS 23. RESULTS: Of the 67 patients, 55(82%) were males and 12(18%) were females. The overall mean age was 25.69±8.305 years. There were 13(19.4%) patients with a history of recurrent disease and previous procedures for pilonidal sinus, while 54(80.6%) had no previous surgery. The median operative time was 35 minutes (interquartile range: 20-45 minutes). Complete wound healing was achieved in 60(89.6%) patients, while recurrence was seen in 7(10.4%). The median time off work was 2.5 days (interquartile range: 1-3 days). Patient satisfaction with the procedure was significantly high (p<0.05). CONCLUSIONS: Endoscopic pilonidal sinus treatment appeared to be a good minimally invasive surgical technique for the treatment of pilonidal sinus disease in terms of both short-term and long-term outcomes.

Hypofractionated proton and carbon ion beam radiotherapy for sacrococcygeal chordoma (ISAC): An open label, randomized, stratified, phase II trial.

INTRODUCTION: Sacrococcygeal chordomas have high recurrence rates and are challenging to treat. METHODS: In this phase II prospective, randomized, stratified trial, the safety and feasibility of hypofractionated ion radiation therapy were investigated. The primary focus was monitored through the incidence of Grade 3-5 NCI-CTC-AE toxicity. Secondary endpoints included local progression-free (LPFS) and overall survival (OS). RESULTS: The study enrolled 82 patients with primary (87 %) and recurrent (13 %) inoperable or incompletely resected sacral chordomas from January 2013 to July 2022, divided equally into proton therapy (Arm A) and carbon ion beam therapy (Arm B) groups, each receiving a total dose of 64 Gy (RBE) in 16 fractions, 5-6 fractions per week. Overall 74 % of patients received no previous surgery and 66 % of tumors were confirmed by a brachyury staining. The mean and median Gross Tumor Volume at the time of treatment (GTV) was 407 ml and 185 ml, respectively. The median follow-up of the surviving patients was 44.7 months, and the 2-year and 4-year OS rates were 96 % and 81 %, respectively. Factors such as smaller GTV and younger age trended towards better OS. The LPFS after 2-year and 4-year was 84 % and 70 %, respectively. Male gender emerged as a significant predictor of LPFS. There was no significant difference between the treatment groups. We observed five grade 4 wound healing disorders (6 %). CONCLUSION: The initial response rates were promising; however local control was not sustained. More comparative research on fractionation schemes is essential to refine treatment approaches for inoperable sacral chordoma.

Coccygectomy for refractory coccydynia, old-fashioned but effective procedure: A retrospective analysis.

PURPOSE: Coccydynia, characterized by persistent pain in the coccygeal region, significantly impacts patients' quality of life. While various treatment modalities exist, including conservative measures and surgical interventions like coccygectomy, optimal management remains unclear. This retrospective cohort study aimed to compare the clinical outcomes, functional improvements, and quality of life in patients with chronic coccydynia undergoing either infiltrative treatment or coccygectomy. METHODS: Data from patients treated at our institution from January 2018 to December 2022 were analyzed. Participants meeting inclusion criteria were divided into two groups: Group A underwent coccygectomy, while Group B received conservative therapy. Clinical assessments, radiographic evaluations, and patient-reported outcomes were collected preoperatively and at follow-up intervals. RESULTS: Of the 223 initially examined patients, 55 met inclusion criteria. Group A (n = 21) underwent coccygectomy, while Group B (n = 34) received conservative therapy. Both groups showed significant pain reduction post-intervention, with sustained improvement in Group A. Functional outcomes favoured Group A, with significant improvements in disability and quality of life measures. Complications were minimal, with only one case of superficial wound infection in Group A. CONCLUSION: Our findings suggest that coccygectomy provides superior and lasting pain relief, functional improvement, and quality of life improvement compared to conservative therapy. While complications were minimal, further research with larger cohorts is warranted to validate these results and explore long-term outcomes. Despite its historical association with complications, advancements in surgical techniques and perioperative care have led to improved outcomes and reduced complication rates. Thus, coccygectomy should be considered in the treatment algorithm for patients with debilitating coccydynia.

Beyond the Tailbone: A Family's Journey Through Caudal Regression Syndrome-A Triple Sibship.

BACKGROUND: Caudal regression syndrome (CRS), also known as caudal agenesis, results from abnormal development of the caudal aspect of the spinal cord and vertebral column due to an earlier abnormality of gastrulation. RESULTS: This report showcases a unique scenario where three siblings, devoid of any prior family history or identifiable risk factors, exhibit symptoms of CRS and receive care at a government-run tertiary facility dedicated to children's health. In establishing a concrete diagnosis, we relied on skeletal surveys, comprehensive symptom evaluation, and medical history assessment. Additionally, we recommended further investigation through magnetic resonance imaging and genetic testing to attain a more in-depth understanding and confirmation of the condition. Unfortunately, the financial constraints faced by the parents led to the unfeasibility of pursuing these advanced diagnostic options. Given the rarity of this syndrome and the limited existing literature, our report is a significant contribution. It marks the first comprehensive exploration of CRS from the genetic and familial predisposition perspective, shedding new light on this rare condition. CONCLUSION: This case series pioneers our understanding of the familial and genetic connections between CRS and sacral agenesis. Strikingly, each subsequent generation has experienced more severe manifestations earlier, furnishing compelling evidence that underpins the genetic predisposition to CRS.

Role of pre-operative endovascular embolization of a giant sacrococcygeal teratoma in neonate: a case report.

BACKGROUND: Giant sacrococcygeal teratomas (SCTs) are at risk of perinatal morbidity and mortality due to their high vascularity. Pre-operative embolization of the feeding arteries, prior to complete surgical resection, may assist in minimizing the intraoperative blood loss by occluding these feeding arteries. CASE PRESENTATION: We present a case of a highly vascular giant SCT in a neonate, which was successfully embolized through an endovascular approach prior to surgery. The femoral artery approach was chosen, with access established using a Micropuncture introducer as a sheath. Embolization was performed using a combination of microcoils, Gelfoam slurry, and polyvinyl alcohol particles. The patient developed femoral artery spasm post-procedure, which resolved with the application of a glyceryl trinitrate patch. CONCLUSIONS: Performing pre-operative endovascular embolization on a giant sacrococcygeal teratoma presents particular challenges, primarily due to the difficulty in assessing small vessels and the potential complications associated with this procedure. Nevertheless, this technique proves exceptionally valuable in helping the surgeon minimize blood loss during surgery, thereby reducing the risks of morbidity and mortality. Comprehensive planning for the embolization procedure is essential, encompassing the identification of potential vascular access points and alternatives, along with careful selection of the appropriate catheter.

Laparoscopic transabdominal-sacrococcygeal approach for resection of Altman type III sacrococcygeal teratoma in adult women: A case report.

INTRODUCTION: Adult sacrococcygeal teratoma (SCT) is a rare disease that is not easily detected or easily missed, and its treatment is based on surgery, including transabdominal, transsacral, or a combination of both, but there are no clear guidelines for diagnosis and treatment. We share a case of Altman type III SCT in order to provide more reference protocols for the diagnosis and treatment of adult SCT, and more importantly to increase our understanding of different types of SCT cases in adults. PATIENT CONCERNS: Our patient was a 31-year-old adult woman who underwent complete surgical resection of a cystic mature teratoma of the right ovary 8 years ago and is currently 13 months postpartum without menstruation, usually with a feeling of anal bulge, with symptoms such as constipation. DIAGNOSIS: We diagnosed SCT by vaginal ultrasonography, computed tomography and magnetic resonance imaging (MRI); benign tumors were considered in the results of serum tumor markers. INTERVENTIONS: We chose the surgical approach of laparoscopic transabdominal-sacrococcygeal approach to completely remove the patient SCT and coccyx. OUTCOMES: The location of SCT is concealed and the clinical symptoms are not obvious. Vaginal ultrasonography, CT and MRI can not only improve the diagnostic rate of SCT, but also understand the size and mass of SCT, providing an exact basis for clinicians to select the laparoscopic transabdominal-sacrococcygeal approach. CONCLUSION: Our sharing increases the reports of rare cases of teratoma with the same histological findings in different organ tissues of the same patient at different times, whether this occurs incidentally requires more case reports and further basic research; in addition, the laparoscopic transabdominal-sacrococcygeal approach is a safe and effective surgical approach for the treatment of Altman type III SCT in adults; finally, this case reminds us that SCT may not affect pregnancy and pregnancy outcomes and provides a reference for the selection of interventions for SCT with pregnancy.

Surgical treatment of sacral pressure injury in ambulating patients during the COVID-19 pandemic: A prospective cohort with complications analysis and elaboration of a surgical treatment protocol.

BACKGROUND: The sudden increase of intensive care unit patients during the coronavirus pandemic led to an increase in the incidence of sacral pressure lesions. Despite being ambulating patients, in many cases the lesions were deep (Grade III and IV), mainly due to the long-term intubation and being bedridden during the pandemic. Most of these wounds necessitated surgical repair. OBJECTIVES: To measure the success and the rate of complications in reconstructions of grade III and IV hospital acquired sacral pressure lesions in ambulating patients after hospitalization for COVID-19. Developing a well-established protocol for surgical treatment of hospital acquired sacral pressure lesions during the COVID-19 pandemic. METHODS: Prospective cohort involving ambulating patients with grades III and IV sacral pressure lesions developed after hospitalization for COVID-19 from May 2020 to August 2020 (4 months). All of them were submitted to reconstruction with fasciocutaneous flaps. Demographics, comorbidities, and preoperative laboratory tests were compared and multivariable-adjusted logistic regression was made in order to identify risk factors for complications. RESULTS: Thirty-eight patients were submitted to fasciocutaneous flaps to repair sacral pressure lesions with a total complication rate of 36.0%. Hemoglobin levels lower than 9.0 mg/dl (p = 0,01), leukocyte levels higher than 11.000/mm3 (p = 0,1), and C Reactive protein levels higher than 142 mg/dl (p = 0,06) at the time of reconstruction and bilateral flaps were independent factors for complications. CONCLUSION: Specific preoperative laboratory tests and surgical techniques were associated with a statistically significant increased complication risk. It was established a protocol for surgical treatment of hospital-acquired sacral pressure lesions to diminish these risks, focusing on ambulating patients during the COVID-19 pandemic.

Effects of alternating pressure patterns on sacral skin blood flow responses in people with spinal cord injury.

Alternating pressure support surface (APSS) is a common support surface for treating pressure injury in individuals with spinal cord injury (SCI). However, conflicting results on the effectiveness of APSS have been reported and may be associated with inappropriate configurations of APSS. The objectives of this study were to compare the different pressure amplitudes (75/5 mmHg [alternating between 75 and 5 mmHg] vs. 65/15 mmHg) and cycle periods (5 min [4 cycles] vs. 2.5 min [8 cycles]) of alternating pressure on sacral skin blood flow responses in 10 individuals with SCI. Sacral skin blood flow during and after loading of four alternating pressure protocols was assessed using laser Doppler flowmetry and was normalised to the value before loading (10-min baseline, 20-min loading and 10-min recovery). The results demonstrated that during the high-pressure phase, there was a significant difference between the 75/5 and 65/15 mmHg protocols (0.3658 ± 0.0688 for 75/5 mmHg and 0.1702 ± 0.0389 for 65/15 mmHg, p < 0.05); and during the low-pressure phase, there was a significant difference between the 75/5 and 65/15 mmHg protocols (1.7184 ± 0.262 for 75/5 mmHg and 0.5916 ± 0.1378 for 65/15 mmHg, p < 0.05). There were no differences between cycle periods in skin blood flow responses. No adverse events were reported. Our finding indicates that the pressure amplitude of alternating pressure is a significant factor affecting sacral skin blood flow responses. An appropriate configuration of alternating pressure is needed to effectively increase skin blood flow and tissue viability in individuals with SCI.

Pulsatile giant sacrococcygeal teratoma appearing like 2nd head.

Sacrococcygeal teratoma is a congenital tumor of early infancy. Eighty percent are present in the first month of life. It is considered to be an extragonadal germ cell tumor that can be diagnosed in utero. It is considered to be a rare tumor in infancy and childhood. Incidences in girls are more common as compared to boys. Association with other congenital abnormalities of the gastrointestinal system, cardiovascular system, and urological manifestations may be present. Association with spinal dysraphism may be present. A complete systemic evaluation needs to be done before any surgical intervention. We hereby present you a case of a 5-day-old male child, born from a non-consanguineous marriage, who presented to us with a large swelling at the coccygeal region. The mass had a variegated consistency with an ear-like appendage over it with associated hair, bones, and necrotic tissue in it. The patient was evaluated with imaging (MRI) which revealed a heterogeneously enhancing mass at the sacrococcygeal region with mild extension into the pre-sacral space suggesting sacrococcygeal teratoma. After a complete evaluation for congenital abnormal abnormalities of other sites, gross total excision of the mass was performed. The post-operative condition of the baby was uneventful. A biopsy of the mass revealed a mature cystic teratoma. The baby is under follow-up at present.