Laparoscopic excision of rare case of recurrent presacral teratoma.

Tumours of the presacral space are rare to present. Most of them are benign masses, very rarely malignant. Surgery is the mainstay of treatment as it establishes the diagnosis and prevents the adverse consequences associated with malignant degeneration and secondary bacterial infection. Their surgical excision is often difficult because of their anatomic location. Very few cases have been reported so far concerning a laparoscopic management of presacral tumour. We hereby present a young girl with recurrent presacral teratoma. She underwent laparoscopic successful excision of tumour with uneventful post-operative recovery. Here, we are highlighting the importance of laparoscopic approach for this scenario in terms additional advantages of minimally invasive approach such as better visualisation of the deep structures in the narrow presacral space, precise dissection in a limited space between the tumour and neighbouring structures with avoiding injury to neurovascular structure.

INTRODUCTION

Presacral tumour refer to a rare and heterogeneous group of lesions that occur in the presacral space, which is a potential space defined superiorly by the rectal peritoneal reflection, anteriorly by the rectum, posteriorly by the sacrum, laterally bound by the ureters, the iliac vessels, the lateral stalks of the rectum and the sacral nerve roots and inferiorly by the presacral fascia.[1] Average incidence of presacral tumour in adult population is 0.0025–0.014. Since multiple embryological structures originates from in presacral space, this area can contain a variety of tumours harbouring diverse histopathology.[2] Teratoma originates from totipotential cells that are abnormally sequestered during in utero development.[3] This case is unique because it demonstrates complete laparoscopic excision of recurrent presacral tumour with uneventful recovery.

CASE REPORT

A 20-year-old girl came with complaints of pain over lower back last 6 months, history of constipation, no history bladder complaints. She had previous history of Robotic-assisted anterior with transcoccygeal posterior (combined approach) excision of presacral tumour 3 years back. Intraoperatively, there were two cystic tumours which are ruptured during dissection, but cyst wall was completely removed. The previous pathological report was ciliated cutaneous cyst. Abdominal examination was unremarkable. There is no evidence of any faecal/urinary incontinence. Local examination healed previous scar over mid sacral area. On per rectal examination, there is extraluminal mass displacing the rectum anteriorly with smooth intact overlying mucosa and rectum loaded with stool. Anal tone was normal. Proctoscopy and per vaginal examination was normal. All laboratory data were within normal limits. Transrectal ultrasound scan shows irregular cystic lesion with solid component of size 6 cm × 4 cm located presacral space with more on the right side [Figure 1a]. Magnetic resonance imaging (MRI) showed a 6 cm × 5 cm × 4 cm size ill-defined, solid cystic lesion noted in presacral region. Posteriorly, the lesion is abutting the lower part of sacrum. Part of lesion has solid component appears hyperintense on T1-weighted and T2-weighted images suggestive fat component which suppressed on fat sat images [Figure 1b]. Sigmoidoscopy revealed no disruption of the mucosa or other abnormalities anywhere. Surgical resection was planned and informed consent was obtained.

Figure 1

(a) Transrectal ultrasound scan: S/o irregular cystic lesion with solid component of size 6 cm × 4 cm located retrorectal more on the right side. (b) Magnetic resonance imaging pelvis: Approximately, 6 cm × 5 cm × 4 cm size ill-defined, solid cystic hyperintese lesion in pre sacral region

A tumour was approached anteriorly with laparoscopy. Patient is in the Trendelenburg position. Ports were placed in the abdomen; 10 mm camera port at 2 cm above umbilicus, 5 mm right hand working port in the right lumbar area, at level of midclavicular line, 5 mm left hand working port in left lumbar area, at the level of the midclavicular line, 5 mm port in left hypochondrium for rectal retraction and another 5 mm port in suprapubic area for adequate exposer operating field. Peritoneum opened in right pararectal region and dissection done in right pararectal and retrorectal space. It was found to be a well-encapsulated cystic lesion, and it was completely free from surrounding structure [Figure 2]. Tumour was resected using a harmonic scalpel after its complete dissection and separation from the surrounding structures with good haemostasis. Specimen retrieved using endobag through left hand working trocar. As previously coccygectomy had been done and tumour appears to benign so removed completely anterior laparoscopically approach. Post-operative recovery was well without any bowel and bladder dysfunction. Operating time was 86 min with 35 ml blood loss. Postoperatively, liquids started after 6 h and diet started the next day. The patient was discharged on a 3rd post-operative day without any complications. The patient is on follow-up last 1 year doing well. The gross appearance an irregular mass of soft tissue measuring 4 cm × 3 cm × 1 cm cut section shows tan soft appearance, light grey coloured, elastic, smooth and unicystic. Microscopically shows fibromuscular tissue with entrapped islands of columnar cells with intestinal type, pancreatic tissue and adipose tissue. There are no immature cells. These findings are consistent with a benign cystic teratoma demonstrating endodermal and mesodermal components.

Figure 2

Laparoscopic image showing tumour in presacral region with adjacent structures

DISCUSSION

Anatomically, the presacral space is the pelvic continuation of the retroperitoneum. The presacral space exists near the confluence of the embryologic hindgut, bony pelvis and neuroectoderm of the spinal cord.[3] Because this ‘potential space’ contains residual totipotent stem cells, a wide variety of neoplasia can develop here, and it can present throughout.[1] Since multiple embryologic structures rise up in presacral space, this area can contain a variety of tumours containing diverse histopathology. Presacral tumours can be classified as congenital, inflammatory, neurogenic, osseous or miscellaneous and each of the above categories is subdivided as benign and malignant lesions. Benign lesions are more common in females whereas malignant tumours have an equivalent distribution.[2] By definition, teratomas contain cellular elements derived from all three germ layers. They originate from totipotential cells that are abnormally sequestered during in utero development. Teratomas are classified as mature, immature, or malignant (teratocarcinoma). About 30% of adult teratomas harbour malignancy at the time of resection.[3] presacral tumours are frequently asymptomatic tumours discovered on routine pelvic or rectal screening investigation is the most common type of presentation.[2] Per rectal examination is simple, but essential for cyst diagnosis because 75% of presacral masses are palpable.[4] Tumour may be initially misdiagnosed as having fistulae-in-ano; pilonidal cysts; perianal abscesses; psychogenic, posttraumatic or postpartum pain; or proctalgia fugax.[3] Incontinence of urine or stool, change in bowel habitus, sensation of inadequate emptying stools, paradoxical diarrhoea are sequelae of the change in the rectal angle due to mass effect.[2] The presence of a postanal skin dimple is may be suggestive of a developmental cyst.[3] Benign lesions frequently silent for a long period. Pain is the most common presenting complaint in infected or malignant cases of tumour.[2]

Transrectal ultrasonography useful in the diagnosis of presacral tumours. Sensitivity of Transrectal ultrasonography is 100% when combined with rigid proctoscopy. Transrectal ultrasonography can predict muscular connection and type of operation. MRI is more beneficial in outlining soft-tissue planes, assessing bony invasion and nerve involvement. Histologic estimation of the tumour can be best achieved using MRI. Biopsy is indicated only when the lesion appears to be unresectable, and a definitive histopathology is required to guide palliative therapy.[2] A transperineal or presacral approach is usually ideal as the biopsy tract falls within the margins of surgical resection.[5]

All surgically fit patients with presacral tumours should be resected, even if they are asymptomatic.[3] Approximately, 30% of resected adult teratoma specimens harbour malignancy.[2] Ludwig and Reynolds have summarised the reasons for operative approach: (1) cystic lesions may become infected, (2) a woman of childbearing age can cause dystocia and prevent vaginal delivery, (3) even benign cystic lesions may harbour occult malignancy, (4) may degenerate to malignancy and (5) resection may improve the quality of life.[3]

The surgical approach depends on location, size of the lesion, its relationship with adjacent structures and planned extent of resection.[4] There are three operative approaches for the resection of presacral tumours are existing; anterior or abdominal, posterior or transsacral and combination of both.[2] The surgeon must avoid sacral nerve injury as bowel and bladder incontinence can occur if damage occurs to the bilateral S3 nerve roots.[1] Anterior approach is suitable for tumours at a higher location having the lowest border above the 4th sacral bone. The absence of sacral involvement is essential for this approach. This approach has advantages of good disclosure of adjacent pelvic structures such as iliac vessels and ureters posterior approach suitable for low-lying tumours which are not extending beyond 4th sacral element combined approach is suitable for the tumour extending both sides of the 4th sacral vertebra. Laparoscopic approach is reported to be feasible and safe.[2] Gonzalez-Crussi et al. found that if coccygectomy not been performed for low laying presacral tumours, recurrence rates were as high as 37%.[6] Overall survival for benign tumours was reported to be nearly 100%. The recurrence rate was reported to be 0%–11% for benign and 47%–75% for malignant tumours.[2]

To conclude, laparoscopic resection for presacral tumour seems safe and feasible with expert's hands. Laparoscopic approach offers additional advantages of minimally invasive approach such as better visualisation of the deep structures in the narrow presacral space, precise dissection in a limited space between the tumour and neighbouring structures with avoiding injury to neurovascular structure along with other such as reduced surgical trauma, less blood loss, with early return to work and excellent cosmetic and also functional outcome.

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Conflicts of interest

There are no conflicts of interest.