Surgical approach of complex cases of uterine fi bromatosis – three case reports and literature review

The main challenges of benign uterine surgery are fertility preservation, choosing the optimal approach (open or minimally invasive) and the management of complications. Conventional laparoscopy is the main technique used for laparoscopic hysterectomy for benign indications. We present three cases of complex patients with uterine fi bromatosis, along with a literature review over the continually regressing limits of the laparoscopic approach of uterine pathology.


Miscarriage and uterine fi broids
First case is a pati ent, 35 years, ferti le (G2P1), with one misscariage 4 months before presentati on, known with uterine fi broma, pelvic pain and menometrorrhagia, who wanted to have another child. She had normal preoperati ve hemoblogin (Hb 12,3g/dL), with negati ve uterine biopsy. Pelvic CT revealed two anterior, subserosal fi broids, 9/6 cm, and 2/1.5 cm respecti vely. Aft er interdisciplinary counsel with the gynecological departament we decided for a laparoscopic procedure. During the procedure, an important bleeding occurred aft er the disecti on of the fi bromas from the uterus. Laparoscopic electrocoagulati on and intracorporeal suturing were not able to achieve haemostasis. We switched to open technique through a Pfannensti el incision, and used separate sutures to stop the bleeding (Fig. 1). Due to the fact that the dissecti on was already made laparoscopically, a small incision of 6 cm was sufi cient to perform a good haemostasis. An incision would have been anyway necessary in order to extract the specimen. The pati ent was discharged aft er a 3 days hospital stay. Postoperati ve pain therapy was similar to the laparoscopic-only procedures. Our approach is always safety fi rst, and in this case safety was maintained without a significantly higher trauma, while the ferti le functi on was preserved in the context of a large fi broma. Aft er the procedure the pati ent had a good quality of life with no dispareunia and no pregnancy during the 6 month follow-up.

Hemorrhagic and septic complications after hysterectomy
Second case is a pati ent, 47 years, who presented with menometrorrhagia with secondary anemia (Hb 9 g/dL), with negati ve uterine biopsy. Pelvic ultrasonography diagnosed a posterior intramural uterine leyomioma of 7/4/6 cm. Laparoscopic hysterectomy was perfomed with no technical diffi culti es in 45 minutes. Culdotomy was made with the 10 mm bipolar electrocoagulati on device (LigaSure). Colporaphy was approached transvaginally, and at the laparoscopic reinspecti on a bleeding from the vaginal stump was observed. Repeated electrocoagulati on was initi ally effi cient with the LigaSure device. Aft er detubati on, 300 mL fresh blood was observed in the drains, and immediate reinterventi on was decided. Due to the fact that laparoscopy was no longer available, a Pfannensti el incision was performed and haemostasis was achieved with intracorporeal suturing of the vaginal stump source. The pati ent was discharged aft er a 3 days hospital stay, in a good conditi on. In the seventh postoperati ve day the pati ent presented for fever, shivers and pelvic pain. The vaginal exam revealed the presence of a small amount of brown, feti d secreti on, with vaginal cuff dehiscence. She was readmitt ed and laboratory fi ndings were signifi cant for leucocytosis (WBC 11,2). Pelvic ultrasonography evidenced a small pelvic collecti on. Reinterventi on through a median incision found vaginal stump necrosis and parti al dehiscence, and methilene blue test for recto-vaginal fi stula was negati ve. The necroti c part of the vagina was resected, and culdoraphy was performed trans-vaginally in healthy ti ssue, due to local infl amati on. Drains were purulent and decreased in fi ve days while a surgical site infecti on was controlled conservati vely. Pelvic pain persist without modifi ed laboratory fi ndings and requiered NSAIDs for seven days, although pelvic CT revealed the absence of any lesion. Excessive electrocoagulati on and low dissecti on of the vagina can damage the vaginal circulati on and cause stump necrosis.

Morbidities associated with uterine leiomyoma
The third pati ent was a post-menopausal woman with recurrent metrorrhagia, secondary anemia (Hb 8.9 g/dl), with negati ve uterine biopsy, with alcoholic cirrhosis (Child A MELD 9) and morbid obesity. At her fi rst admission, pelvi-abdominal CT revealed a large polyfi broid uterus (25/18 cm), along with portal hypertension, splenomegalia and macronodular liver. She also presented severe superfi cial thrombofl ebiti s. Anti coagulati on medicati on, along with NSAIDs and supporti ve management (extremity elevati on, cool compresses and class II compression stockings) was initi ated. Surgery was postponed unti l complete resoluti on of the thrombofl ebiti s.
Aft er a month, the pati ent presented for acute uterine bleeding (Hb 7.8 g/dl). Emergency total histerectomy with bilateral anexectomy was performed, initi ated laparoscopically but converted due to large uterine varicose veins (Fig. 2). Aft er conversion, the procedure was uneventf ul. In the fi rst postoperati ve day, the pati ent had serous drains with 1,000 mL and leukocytosis (WBC 28.000/mmc), and intravenous ureterography revealed grade II hydronephrosis, without contrast leakage. Ureteroscopy found no ureteral lesion, but right J-J catheter was inserted for a possible extrinsec compression. Liver support with diureti cs, sodium restricti on and albumin limited the asciti s, with the extracti on of the drains aft er 16 days. Pathology showed an unexpected Krukenberg tumor on the left ovary, macroscopically normal, and polyfi bromatous uterus. Upper endoscopy was oncologically insignifi cant, and found esophageal varicose veins grade II and portal gastropathy. At the same ti me, colonoscopy visualized an ulcerati ve caecal tumor, without stenosis. The pati ent was discussed in our tumor board meeti ng and surgical fi rst approach was decided. Right hemicolectomy with ileo-transverse-anastomosis was performed. Despite the intensive pre-and postoperati ve care and sup- porti ve measures, the pati ent developed liver failure from the fi rst postoperati ve day with hepato-renal syndrome, with asciti s, hemodynamic complicati ons due to low sistemic vascular resistance, modifi ed coagulati on parameter (INR 2.61). In the 4th postoperati ve day, anastomoti c leakage with local peritoniti s also occurred. Ileostomy and intensive support proved insuffi cient to treat MODS, and the pati ent died the fi rst day aft er ileostomy.

DISCUSSIONS Fibroids and sterility / miscarriage
Fibroids are the most common benign tumours of the female genital tract and are associated with numerous clinical problems, including a possible negati ve impact on ferti lity. The reported incidence of fi broids in pregnancy ranges from 0.1-10.7% of all pregnancies (1,2). Women who had a miscarriage experienced less fi broid regression (3), so the discovery of a uterine myoma needs to consider the responsibility of myomas in inferti lity, but also its impact on a future pregnancy. Fibroids represent one of the most frequent indicati ons for major surgery in premenopausal women and as such, they constitute a major public health cost (4).
In women requesti ng preservati on of ferti lity, fi broids can be surgically removed (myomectomy) by laparotomy, laparoscopically or hysteroscopically depending on the size, site and type of fi broid (5). Submucous myoma should be treated by a hysteroscopic approach. Intramural and subserous myomas in women who opt for nonsurgical treatment could be treated with uterine artery embolizati on (UAE), high-intensity focused ultrasound (HIFU), or medical treatment. All interventi ons aside from hysterectomy provide temporary relief, although myomectomy, UAE, and HIFU provides more durable symptom relief relati ve to current medical management. Pati ents wishing to preserve their ferti lity and presenti ng with subserous leyomioma are best treated by myomectomy, which can be done by laparoscopy (6).
Myomectomy is however a procedure that is not without risk and can result in serious complicati ons. It is therefore essenti al to determine whether such a procedure can result in an improvement in ferti lity and, if so, to then determine the ideal surgical approach. Informed consent must include a clear explanati on of the possibility of conversion and even total hysterectomy. As previous presented in our case, even in case of conversion aft er laparoscopic dissecti on a smooth postoperati ve course was observed.

Laparoscopic myomectomy
Few data address the opti mal criteria for a successful laparoscopic myomectomy, and the ability to successfully perform a laparoscopic myomectomy also depends in large part on surgical experti se. The largest study was a prospecti ve multi center study of 2050 women undergoing laparoscopic myomectomy (7). Leiomyoma characteristi cs that were signifi cantly associated with major complicati ons (eg, bleeding requiring blood transfusion, visceral injury, procedural failure) included: size of myoma >5 cm; >3 myomas removed; and intraligamentous locati on; intramural myomas were signifi cantly associated with an increase in minor, but not major, complicati ons (eg, fever, uterine manipulator injuries).
Laparoscopic myomectomy is a procedure associated with less subjecti vely reported postoperati ve pain, lower postoperati ve fever and shorter hospital stay compared with all types of open myomectomy. No evidence suggested a diff erence in recurrence risk between laparoscopic and open myomectomy. More studies are needed to assess rates of uterine rupture, occurrence of thromboembolism, need for repeat myomectomy and hysterectomy at a later stage (8).
Myomas are also associated with an increased rate of obstetric complicati ons, as adhesions are the main complicati on of myomectomy. Pregnancy rate in cases of myomectomy by laparotomy and laparoscopy is similar (9). Regarding the surgical approach to myomecto- my, current evidence from two randomised controlled trials suggests there is no signifi cant difference between the laparoscopic and open approach regarding ferti lity performance. This evidence needs to be viewed with cauti on due to the small number of studies (5). Postoperati ve pain is subjecti vely reported as lower in laparoscopic myomecyomies in comparison to open procedures (8).

Pfannenstiel incision -the handy approach
We used Pfannensti el incisions for intact specimen extracti on, but also for haemostati c reinterventi on aft er a laparoscopic approach of the uterus. At the same ti me, specimen extracti on through a Pfannensti el incision has bett er outcome than expanded port site incisions. Pfannensti el incision has less morbidity, pain score, and hospital stay, although both incisions are associated with high operati ve sati sfacti on, good cosmesis, and a low rate of wound complicati ons (10). The rate of incisional hernia aft er a Pfannensti el incision is the lowest when compared to minilaparotomy and standard laparotomy and should be the incision of choice for hand assistance and specimen extracti on in minimally invasive procedures wherever applicable (11).

Complications in laparoscopic hysterectomies
In our department we try to perform laparoscopic hysterectomies as oft en as possible. The technological development, but essenti ally the experience gained by surgeons are the key factors in accepti ng and promulgati ng the technique (12).
Secondary hemorrhage is rare but may occur more oft en aft er total laparoscopic hysterectomy than aft er other hysterectomy approaches. Whether it is related to the applicati on of thermal energy to ti ssues, which causes more ti ssue necrosis and devascularizati on than sharp culdotomy in abdominal and vaginal hysterectomies, is not clear. A large uterus size, excessive use of an energy source for the uterine artery and for culdotomy may play a role (17).
Vaginal cuff necrosis without hemorrhage is another independent consequence of excessive electrocoagulati on and it can be clinically confused with recto-vaginal fi stula. Vaginal cuff dehiscence is an uncommon complicati on, but the incidence is highest following a laparoscopic procedure. As an example, a retrospecti ve study of over 12,000 hysterectomies reported the following rates of vaginal cuff dehiscence: abdominal (0.38%), vaginal (0.11%), and laparoscopic (0.75%) (18).
Evaluati on of postoperati ve complicati ons might be bett er assesed using Clavien-Dindo classifi cati on and we suggest introducti on of CD classifi cati on for comparability of uterine procedures. So far, in this area it was only used for the evaluati on of pelvic prolapse surgery (19). In this paper we present three cases where all fi ve Clavien classes of complicati ons occurred.

Associated morbidities in fi broids and fi rst lesion mirage
Associati on of comorbiditi es such as morbid obesity, macronodular cirrhosis with portal hypertension are negati ve prognosti c factors for the hysterectomy outcome. Menstrual disorders and menorrhagia in menopausal women might be a consequence of high concentrati on of oestrogens due to cirrhosis, as the main indicati on for hysterectomy among pati ents with cirrhosis is menorrhagia (20,21).
Emergency hysterectomy can mask other pathologies, such as malignancies, encountered in the third presented case. Without doubti ng the fi rst lesion mirage, we wouldn't follow another clinical course in the forementi oned pati ents.

CONCLUSIONS
Laparoscopic hysterectomy is nowdays a standardized procedure. However, indicati ons and perioperati ve morbiditi es sti ll have disputable areas.
In a center with high load of laparoscopic pelvic procedures, in all three cases the intraoperati ve and postoperati ve complicati ons were unexpected and really challenging for experienced surgeons. Team work and interdisciplinary communicati on, as well as safety fi rst approach, were the key factors for successful management of our pati ents. However, against all odds, there are situati ons when the experience, cauti on and intensive care are simply not enough for a positi ve outcome.