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1 From Early Pioneers to the PelvEx Collaborative

       Éanna J. Ryan1 and P. Ronan O’Connell1,2

       1 Department of Surgery, St. Vincent’s University Hospital, Dublin, Ireland

       2 Royal College of Surgeons in Ireland, Dublin, Ireland

Background

      Pelvic exenteration, involving radical multivisceral resection of the pelvic organs, represents the best treatment option. The first report of pelvic exenteration was in 1948 by Alexander Brunschwig of the Memorial Hospital (New York USA), as a palliative procedure for cervical cancer [1]. Due to high morbidity and mortality rates many considered palliative exenteration too radical, and it was performed only in a small number of centers in North America [2].

      Technologic advancements, surgical innovations, and improved perioperative care facilitated the evolution of safer and more radical exenterative techniques for the treatment of advanced gastrointestinal and urogynecological malignancies [3]. Worldwide collaborative data [4, 5] have demonstrated that a negative resection margin is crucial in predicting survival and quality of life after surgery. Carefully selected patients who undergo en‐bloc resection of contiguously involved anatomic structures with R0 resection margins can expect good long‐term survival with acceptable levels of morbidity [4, 5].

The Pioneers

      Eugene M. Bricker (Columbia, USA), a contemporary of Brunschwig, had been independently performing exenterative procedures beginning in 1940 [6]. Due to adverse outcomes and the interruption of World War II, his experience remained unpublished [6]. Jesse E. Thompson (Dallas, USA), one of the founders of vascular surgery as a subspecialty, and Chester W. Howe (Boston, USA) reported the first case of “complete pelvic evisceration” for locally advanced rectal cancer (LARC) in 1950. Other early advocates of the concept included Lyon H. Appleby (Vancouver, Canada), who performed a procedure he termed a “proctocystectomy” [7], and Edgar S. Brintnall (a general and vascular surgeon) and Rubin H. Flocks (an early urologist from Iowa, USA), who termed their procedure “pelvic viscerectomy” [8].

      Brunschwig’s Operation

While elsewhere PE was being developed principally for patients with LARC, in New York, Alexander Brunschwig was performing PE as a palliative procedure for locally advanced gynecologic malignancies. Before the introduction of PE, the prognosis for locally advanced cervical cancer was particularly poor. External beam radiation therapy was the mainstay of management. Local extension commonly occurred and cure rates were as low as 20% for primary disease [9]. Forty percent of deaths were the result of advanced disease confined to the pelvis [10]. Patients with end‐stage malignancy suffered refractory pain, as well as intestinal and ureteric obstruction as major complications [11, 12].

Brunschwig, who had been among the first to report a one‐stage radical pancreatico‐duodenectomy in 1937 [1, 13], observed that PE was a “procedure of desperation since all other attempts to control the disease had failed.” Initially his only selection criterion was that disease must be “confined to the pelvis.” Interestingly, “not a single patient refused the operation even after detailed explanation of the procedure and the complications associated with surgery” [1]. The operative approach was similair (Figure 1.1).

      Although Many surgeons were critical, considering it “a thoughtless form of mutilation, with limited chance of success for palliation, much less cure” [14]. In the earliest series, the survival outcomes were poor, with one in every three operations resulting in perioperative mortality [1, 15]. In Brunschwig’s 1948 article, he reported operating on 22 patients with 5 deaths. [4].

      By 1950, Bricker was also investigating the role of PE in the management of cervical cancer. His first patient, despite widespread local invasion, had a disease‐free survival of 42 years [6]. The suitability of PE for the management of cervical and other gynecological cancers was later confirmed by Brunschwig in several series [16, 17]. In the ensuing decades, several units (mostly in North America) increasingly performed PE for advanced cancer of the vulva [18], ovary [19], and prostate [20], and for pelvic sarcoma [21]. The first documented non‐malignant application for PE was for management of severe radiation necrosis of several pelvic organs in 1951. This remained a relatively common indication for PE until more contemporary radiation therapies became available [22].

      Evolution in Pelvic Exenterative Surgery

      Urinary Reconstruction

      The key challenge in extended pelvic resection was urinary tract reconstruction. Though urinary diversion techniques had been described since 1852, leakage and infection issues resulted in many modifications in technique over the last century [23]. In 1909, Verhoogan and De Graeuwe (Brussels, Belgium) implanted ureters into an isolated segment of terminal ileum draining via an appendicostomy [24]. However, isolated ileal segments temporarily fell out of use [25]. Over the next three decades, Robert C. Coffey (Oregan, USA) experimented with various methods of bladder substitution by implanting ureters into the residual colon [26, 27]. Although he presented his outcomes outcomes in 1925 they were never published because “exposure of the ureters and kidneys to the fecal stream often led to sepsis, hyperchloremic acidosis, and kidney failure” [24]. Brunschwig’s favored technique of “wet colostomy” was essentially reproduction of Coffey’s method and suffered from the same shortcomings [22].

Other pioneers interested in this type of surgery had also attempted the creation of artificial bladders from bowel or alternatively developing cutaneous ureterostomies [22]. Appleby (Vancouver, Canada) examined the possibility of transferring both ureters to an intact cecum draining through a sigmoid colostomy, but with limited effect [7]. Similarly, Bricker created a diversion that involved isolation of a cecal segment “to be drained intermittently of urine through a catheter” [6]. Gilchrist and colleagues reported attaining successful continence with the construction of an intra‐abdominal reservoir from isolated cecum draining via the terminal ileum [28]. However, Bricker was unable to duplicate these results and chronic leakage of urine frustrated clinicians and patients alike (Figure 1.2) [29].