Surgical strategy for presacral tumors: analysis of 33 cases

4086Chin Med J 2011;124(23):4086-4091Clinical experienceSurgical strategy for presacral tumors: analysis of 33 casesLI Guo dong, CHEN Kai, FU Dong, MA Xiao-jun, SUN Meng-xiong, SUN Wei and CAI Zheng-dongKeywords: presacral tumors; surgical strategy; diagnosisBackground Presacral tumors are highly infrequent tumors located in the space known as presacral or retrorectalspace. Although there have been substantial improvements in the prognosis of patients with malignant presacral tumors,the development of newer surgical strategy is likely to further improve the oncologic outcomes of malignant presacraltumors. The aim of this article was to report our experience in 33 cases, and to review the surgical stategy, pathologicalfeatures and the prevention of complications from our experience.Methods A retrospective analysis was conducted on 33 cases (20 male and 13 female) with presacral tumors surgicallytreated in our hospital between January 1998 and April 2009. The surgical approaches included trans-abdominal in 10cases (30%), trans-sacral in 18 cases (55%) and combined abdominal-sacral in 5 cases (15%). AII patients got followedup (14 123 months, mean of 45.1 months). At last, the general information, clinical symptoms, histodiagnosis, surgicaltypes and postoperative complications of all cases in our series were assessed.Results Ages of 33 patients ranged from 18 to 71 years, with an average of 48.5 years. Pathological findings: 6epidermoid cysts, 5 teratomas, 3 leiomyomas, 9 neurofibromas, 5 neurilemmomas, 1 enterogenous cyst, 1 liposarcoma,1 leiomyosarcoma, 1 angiosarcoma, and 1 neurofibrosarcoma. All tumors were excised with no perioperative death. Acolostomy was taken in one case with angiosarcoma involving the rectum because of the intraoperative injury of therectum. Blood loss during surgery was 400 -11000 ml (mean of 2400 ml). Four (12%) cases had local recurrence duringfollow-up: 2 because of inadequate drainage ater dermoidectomy, both of them were cured by surgical resection anddrainage; recurrence occurred in a case of teratoma in 18 months ater surgey, cured by a trans-sacral excision; localrecurrence and lung metastasis occurred simultaneously in a case of angiosarcoma in 6 months postoperatively and thepatient died one month later of respiratory failure.Conclusions The main treatment of most presacral tumors is surgical resection. Selection of surgical approach is veryimportant for complete resection of the presacral tumors. The location, size and peculiarities of tumors, conditions of theskin and soft tissues and the patients' somatotype are all determinative factors. Multidisciplinary cooperation is also verynecessary.Chin Med J 2011;124(23):4086- 4091Dresacral tumors are highly infrequent conditions.Departments of Orthopedics, General Surgery, andAlthough the true incidence of such tumors isGynecology respectively in 10th People's Hospital ofunknown, it is estimated at one in every 40 000 hospitalShanghai, China. The first symptom was variable,admissions.2 They are usually benign and located in theincluding abdominal mass in 9 (27%)， defecationarea known as the presacral or retrorectal space. Thedisability in 6 (18%)， diarrhea in 2 (6%)， urinarytumors may derive from remains of embryologicalsymptoms (urinaryfrequency, urgent micturition,structures or may be a consequence of malignization indysuresia, and uroclepsia) in 3 (9%), sacrococcygealthe area.' In general, they are asymptomatic or haveregion pain in 3 (9%), lower extremity pain in 3 (9%) andunspecific symptomatology, making their diagnosislow back pain in 2 (6%), the other 5 cases (15%) withoutdifficult and on occasion by chance. The fact that the areaany symptoms were diagnosed by chance during a routinewhere they reside is asymptomatically occult and diffcultdigital rectal examination. The mean duration fromto access makes experience in surgical intervention abeginning of symptoms to diagnoses was 5.5 monthsrequisite for treating them.4 A good knowledge of its(1 week-1 year) (Table 1). The locations and circum-peculiarities is also important. The aim of this study wasto discuss the surgical strategy and pathological featuresDOL: 10.3760/cmaj.issn.0366 6999.201 1.23.044Department of Orthopaedics, Shanghai Tenth People's Hospital,from our experience of surgical treatment with 3Tongji University School of Medicine, Shanghai 200072, Chinaoperated cases.(LiGD,ChenK,FuD,MaXJ,SunMX,SunwandCaiZD)Correspondence to: Dr. CAI Zheng-dong,_ Department ofMETHODSOrthopaedics,Tongji UniversitySchool of中国煤化工72,China (Te:Patients86-21-56305CNMHGm)This researct:CYHun b, gians HIUI umunghai Natural ScienceFrom January 1998 to April 2009, 33 patients of presacralFund (No. 1IZR1428400), Shanghai Municipal HealthBureautumors (20 male and 13 female) with a median age ofFund (No. 2010163) and National Ministry of Education Fund48.5 years (18 -71 years) were surgically treated in the(No. 20100072120056)..4088Chin Med J 201;124(23):4086-4091exposed, digital rectal examination should be made againadjunctive therapy, and etc.to confirm the tumor extention. The dissociation ofpresacral tumor often started from distal margin and thenStatistical analysisthe bilateral margin. During this procedure, it wasBlood loss was analyzed with unpaired t test by the Stataimportant to make sure that sphincter and posterior rectal11 (College Station, Texas 77845, USA). A P value lesswall were protected. Attention should be paid to thethan 0.05 was considered statistically significant.possibility of presacral vessels bleeding when separatingthe cephalic margin. The key to dissociate anterior sacralRESULTSarea was pulling-down the tumor with left hand as toform a tension and then to find the presacral space usingWe adopted trans-sacral approach in 18 patients, simpleforceps. It is better to use ultrasound knife or APC (Argontrans- abdominal approach in 10 patients and 5 combinedPlasma Coagulation) to cut off the cephalic margin theabdominal-sacral cases.tumor. A vacuum drainage device should be placed in thecavity left before closing the incision eventually.Pathological types were listed as follows: epidermoid cyst,teratoma,leiomyoma,neurofibroma,neurinoma,Trans-abdominal approachenterogenous cyst, liposarcoma, leiomyosarcoma,Criterion for this approach was that the tumor was biggerangiosarcoma, neurofibrosarcoma, and etc. (Table 2).and located over S3 when it was difficult to explore usingtranssacral method. The median abdominal incision wasTable 2. Pathological resultsmade at inferior belly and then dissected the retrorectalItemsBenign29(87.8)space according to tumor location at a lithotomy position.The key point during isolating was protecting rectalEpidermoid cyst6(18.1)vessels and mesorectum. It was suggested that presacralTeratoma5(15.1)Leiomyoma3 (9.1)space should be found prior to dissociating the retum andNeurofibroma9(27.3)the tumor in order to prevent hemorrage of presacralSchwannoma5(15.2)venous plexus (PSVP) which was usually lead to by usingEnterogenous cyst1(3.0)Malignant4(12.1)electrotome blindly. Appropriate drainage method wasLiposarcoma1 (3.0)applied depending on the wound,LeiomyosarcomaAngiosarcomaCombined approachNeurofibrosarcomaFor those cases with distal margin lower than S3 and thecephalic margin higher, it is not possible to remove theNo perioperative death occurred. All tumors weretumor only by trans sacral or trans- abdominal approach.completely resected. Blood loss during surgery wasThe tumor can be entirely resected by the combination of400-11000 ml (mean, 2400 ml). The value othe two approaches above. Benign presacral tumors couldinteroperative blood loss showed a significant differenceget clinical cure with radical excision, while for(t=2.8452, P=0.0078) between the 13 cases before 2006malignant ones local adjunctive radiotherapy had been an(mean, 3646 ml) and the 20 cases after 2006 (mean, 1595essential part.n)， which means 24 hours is the optimal intervalbetween selective artery embolization and surgery ratherNo matter what kind of approach chosen, intraoperativethan 72 hours.digital rectal examination is vital to avoid rectum injurywhile dissociating the tumor. For trans-sacral anUncontrolled presacral phleborrhagia occurred in 2 casescombined approaches, at least unilateral S3 and all of the(I leiomyoma, 1 neurofbroma) intraoperatively. Both gotS1-S2 nerve roots should be reserved and protected.gauze packing during surgery. The gauze was pulled out 1week after operation. A colostomy was taken in one casePostoperative treatmentswith angiosarcoma involving the rectum because of theThree cases with malignant tumors (1 liposarcoma, 1intraoperative injury of the rectum. There were 23leiomyosarcoma and1 angiosarcoma) got localtrans-sacral cases (18 of simple trans-sacral approach, 5radiotherapy (45 Gy) 4 weeks after surgery.of combined approach). Five (22%) of the 23 cases hadwound complications postoperative: incision marginFollow-upnecrosis and disunion in 3 cases (2 of them got localFollow-up was done 1, 2, 3, 6 and 12 months afterdebridement, drainage and secondary suture); 1surgery by clinic service or telephone. During the secondpostradiotherapy case got local flaps transfer because ofyear, follow-up was done every 3 months. And from thelarge cutaneous deficiencv: 1 case nf skin and superficialthird year, follow-up was donehalf-yearly. Routinefascia infecti中国煤化工debridement andworkflow includes X-ray and CT scan of thelocal flaps tralYHCNM HGsacrococcygeal region as well as the evaluation of healingof the local wound, recurrence, functions of defecation,All patients in our series got followed up (14 -123 months,sensation and muscle strength of lower limbs, effect ofmean, 45.1 months). Four (12%) cases had localChinese Medical Journal 2011;124(23):4086 40914089recurrence during follow-up: 2 cases because ofnot in favor generally7 because histodiagnosis rarelyinadequate drainage after dermoidectomy, both of themaffects the decision of operation.were cured by surgical ablation and drainage; recurrenceoccurred in a case of teratoma in 18 months after surgery,Pathological featurescured by a trans-sacral excision; local recurrence and lungThe heterogeneity of histology leadsmultiplemetastasis occurred simultaneously in a case ofclassifications of presacral tumors: (1) congenital tumorsangiosarcoma in 6 months postoperatively and the patient(including dermoid/epidermoid cyst, teratoma, chordoma,diedd one month later of respiratory failure. Four (12%)cases were found with symptoms of sphincter dysfunctionneurofibrosarcoma, neurinoma, and etc); (3) inflamsuch as disability of defecation. They all neededgoiters (including abnormal granuloma, rectal sinusesadjunctive medication. All of the 4 cases were benignabscess, andetc.); (4) osseous neoplasms (includingneurogenic tumors, including 1 neurofibroma and 3osteoma, osteochondroma, osteoblastic sarcoma, and etc.);neurinomas.and (5) others (including lipoma, liposarcoma, fibroma,fibrosarcoma, and etC.).DISCUSSIONSome reportseven mentioned extremely exceptionalPresacral tumors are tumors of the presacral region whichpresacral neoplasms such as neuroendocrine tumors. Inis constrained posteriorly by the sacrum and coccyx andthe 33 cases of our series, neurogenic tumors (15 cases,the rectum anteriorly.Generally its upper margin is below46%) are the most common types. Next to them arethe S2 level, and sometimes higher than the SI level,congenital epidermoid cyst (6 cases, 18%) and teratomaeven up to the L5 vertebral level. The presacral region is(5 cases, 15%). Most reports show that benign presacralbound by the rectum in the front, the presacral venoustumors are more than malignant ones. In our series, 29plexus and sacral nerve roots from behind, and the ureterscases (88%) are benign, and only 4 cases (12%)and iliac vessels laterally. As the area where the tumorsmalignant. But there are still some reports, showing thatreside is asymptomatically occult and difficult to access,48% of presacral neoplasms are malignant.'misdiagnoses are common. Owing to this, whendiagnosed, the tumor is huge mostly (in our series theSurgerydiameter ranges 2.4 -21.4 cm, mean 9.3 cm). And theSome of presacral tumors are malignant; congenitalnarrow space of the lesser pelvis and the anatomicalteratoma has degeneration tendency; the cystic lesionscomplexity of the presacral region bring great difficultiesmay have the risk of infection. So, all presacral tumors/to surgical treatment of presacral neoplasms. Negligencelesions should be considered for surgical resection exceptin operation may cause serious complications, such asthose who have absolute surgical contraindications.- Andhemorhage of the presacral venous plexus and theboth benign and malignant presacral tumors should beinjuries of rectum or the sacral nerve roots. Surgicalconsidered marginally resected in order to avoid localmanagement should usually be determined by therecurrence.cooperationamong orthopedics, general surgical,urological, gynaecological surgeons because of thecomplex anatomy of this region. The presacral tumorsthe presacral tumors are usually extremely close to thehave been challenging to surgeons for a long time.rectum and the anal canal. What's more, for some patientswith huge presacral tumors, ureteral catheter should beDiagnosisprepared to protect the ureters from intraoperativehe symptoms are often insidious and early diagnosis isinjuries.dificult when presacral tumors are small. Diagnosis isoften made when the tumor is big enough to cause overtThe surgical approach of these tumors may depend onsymptoms or during health examination. Digital rectalvarious factors. Tumor size, location, peculiarity, patients'examination is very important for diagnosis of presacralsomatotype and conditions of local skin and soft tissuetumors.' Testini et al reported that 76%- -90% of patients(with or without radiotherapy, etc.) are all decisive factors.presented with palpable tumors in the digital rectalSuitable approach, which is the key to succesfulexamination. And in our series, the rate is 82% (27/33).operation, may help provide an optimal exposure, reduceAnother great value of digital rectal examinationdamage and complications. We recommend threeaccording to our experience is that if we could touch theapproaches mainly according to our experience:superior margin of tumor, we may choose trans-sacraltrans-abdominal approach, trans-sacral approach andapproach during operation. Though there are manycombined abdominal-sacral approach. Trans-abdominalauxiliary examinations such as endoscope and abdominalapproach istumors above S3中国煤化工ultrasound, CT and MRI are most widely used imaginglevel. The aated: we firstlytechniques.' Because they are more accurate and moremobilize theTYHCNM H Gto the front, andvaluable to help us choose suitable surgical methods. Allthen excise the tumor. Great attention should be paid topresacral tumors should be considered for surgicalpresacral hemorrhage because the middle sacral bloodresection when diagnosed. Preoperative local biopsy isvessels and the presacral venous plexus are in this region.4090Chin Med J 2011;124(23):4086- 4091Every blood vessel should be ligated carefully whenand gentle operations are keys to the success of thefreeing the tumor. Measures also should be taken topresacral surgery.protect the ureter and main nervous branches in thepresacral region. Trans-sacral approach is applied toMultidisciplinary cooperationtumors below S3 level, or those which are not large andBecause of the anatomical complexity of the conditionwhose superior margin can be reached by digital rectal(intestines, genitourinary organs, iliac vessels, and etc.)examination. 4 This approach was adopted in 18 casescooperations among orthopedics， general surgery,of our series. Compared with trans-abdominal approach,urological, and gynaecological departments are extremelytrans-sacral approach is more direct and morenecessary. In our series, surgeons of different relevantcommodious to adopt. The operating field of the tumor isdepartments participated in the preoperative discussionclearer, while the adverse influences of intestines areroutinely. For patients with large presacral tumors withavoided, so patients may get quicker recovery afterpreoperative intravenous pyelography showed theoperations." Combined abdominal-sacral approach iscompressed ureter, urological surgeons were consulted touseful for excessively large presacral tumors which couldhelp to place the ureteral catheter before the surgery. Ifnot be excised by, trans-abdominal or trans-sacralthe tumor had infiltrated uterus and appendants, theapproach separately.' First we dissociate tumor bodygynaecologists were consulted to help in the operation. Iffrom upper posterior rectum and then close the abdominalpreoperative CTMRI showed that it was hard towall, then switch to the prone position and excise thedissociate the tumor from the rectum wall, the generaltumor through trans-sacral approach. The greatestsurgeons were consulted to help intraoperative. If DSAadvantage of this approach is that it can reduce theshowed that the tumor is highly vascularized and hasincidence of organ injuries as well as resect the tumorobvious compression of the iliac vessels, the vascularcompletely. In our series, 5 cases with huge presacralsurgeons were consulted to assist in the surgery routinelytumors were successful resected by this combinedin our series. Multidisciplinary cooperation helps to raiseapproach.the resection rate of the tumors, avoid unnecessaryinjuries and improve the prognosis and life quality of theNo matter what approach is chosen, surgical treatment forpatient. For some rare presacralmalignancies,presacral tumors is highly risky of severe complications.cooperation among orthopedic surgeons, oncologists andHemorrhage of the presacral venous plexus, injuries ofradiotherapist is also very important.rectus and sacral nerves are the most frequent and seriouscomplications of presacral operations. Hemorrhage of theREFERENCESpresacral venous plexus is the main cause ofintraoperative death while common hemostasis methods1. Whittaker LD, Pemberton JD. Tumors ventral to the sacrum.have little effects." 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