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OPEN ACCESS
Jacobs Journal of Nephrology and Urology
Systematic Employment of Laparoscopic Decortication for Maximal
Pain Control in Polycystic Kidney DiseaseNeetika Garg, MD1*, Andrew
A. Wagner, MD2, Theodore I. Steinman, MD1 1Nephrology Division,
Department of Medicine, Beth Israel Deaconess Medical
Center2Urology Division, Department of Surgery, Beth Israel
Deaconess Medical Center
*Corresponding author: Neetika Garg, Beth Israel Deaconess
Medical Center/ Harvard Medical School, 185 Pilgrim Road/ Farr
8,
Boston, MA 02215, Tel: 617-632-9880; Fax: 617-632-9890; Email:
[email protected]
Received: 07-31-2014
Accepted: 09-22-2014
Published: 10-12-2014
Copyright: 2014 Neetika
Case Series
Cite this article: Garg N. Systematic Employment of Laparoscopic
Decortication for Maximal Pain Control in Polycystic Kidney
Disease. J J Nephro Urol. 2014, 1(2): 11.
Abstract
More than 60 percent of patients with Autosomal Dominant
Polycystic Kidney Disease (ADPKD) experience chronic ab-dominal or
flank pain over the course of their lifetime. Individual enlarging
cysts can cause pain through stretching the renal capsule and/or
compression of surrounding structures. This pain is most often
localized to the anterior abdomen. The usual strategy with
laparoscopic cyst decortication (LCD) has been to excise/unroof as
many cysts close to the surface of the kidney as possible. However,
this provides lasting relief in only 50 to 60 percent of the
treated patients are associated with prolonged operative times, and
causes significant post-operative complications such as urinary
leaks leading to formation of urinomas. Of particular concern is
the potentially detrimental effect of the procedure on renal
function. Here, we describe a systematic approach for employing
this technique for cyst related pain in ADPKD: 1) Patient must be
able to provide single finger pinpoint location of chronic
reproducible pain lasting at least 6 months. This pain must be
consistently moderate to severe in intensity (more than 5 on a
10-point visual analog scale) despite maximal conservative therapy
not including chronic opioid analgesic use. 2) The area of maximal
pain/ tenderness is identified using a skin marker. 3)
Ultrasonography is used to identify culprit cysts that are at least
4 cm in size and are located directly underneath the marked skin
area. 4) Our team urologist experienced in surgical management of
ADPKD employs the LCD technique, never unroofing more than three
cysts. We document successful use of this approach achieving
excellent and lasting pain control, while minimizing op-erative
times and other adverse effects in three patients who underwent a
total of four procedures.
Keywords
Laparoscopic Cyst Decortication; Marsupialization; Unroofing;
Polycystic Kidney Disease; PKD; Cyst Pain; Chronic Pain; Pain
Management; Review
Abbreviations
ADPKD: Autosomal Dominant Polycystic Kidney Disease; LCD:
Laparoscopic Cyst Decortication; Cr Cl: Creatinine Clearance; VAS:
Visual Analog Scale;MRI: Magnetic Resonance Imaging;
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Jacobs Publishers 2
Cite this article: Garg N. Systematic Employment of Laparoscopic
Decortication for Maximal Pain Control in Polycystic Kidney
Disease. J J Nephro Urol. 2014, 1(2): 11.
USG: Ultrasonography;CKD: Chronic Kidney Disease;eGFR: Estimated
Glomerular Filtration Rate;JP: Jackson-Pratt; POD: Post-Operative
Day;
Introduction
Autosomal Dominant Polycystic Kidney Disease (ADPKD) is the most
common inherited cystic renal disease; it affects 1 in every 400 to
1000 individuals and is the underlying etiology in approximately 5%
of the patients who initiate dialysis every year in the United
States. Pain is a common early symptom in the course of ADPKD. More
than 60 percent of the patients are affected by abdominal and/or
flank pain in their lifetime, often decades before renal
insufficiency sets in [1,2]. Additionally, chronic pain is a
complaint in almost half the patients even be-fore the diagnosis is
established and represents the most com-mon symptom leading to the
diagnosis [3].
Renal cysts cause two distinct types of pain in ADPKD:
1) Pain due to overall increased renal mass: Enlarging cysts
cause an increase in abdominal girth that, over time, leads to
progressive maladaptive postural changes, including an exaggerated
pelvic tilt and increased lumbar lordosis [4]. Resultant mechanical
strain on the lumbosacral muscles ulti-mately leads to hypertrophy
of these muscles and progressive degenerative spine disease. In a
personal observation by one of the authors (TIS), in 10 patients
with ADPKD average lum-bodorsal muscle thickness at L4-S1 level on
abdominal MRI done to assess cyst volume was 38.7 mm, in contrast
to 31.3 mm in ten matched individuals without ADPKD undergoing MRI
for unrelated reasons [4]. Typical presentation is chronic back
pain that is usually made worse by standing or walking.
Non-pharmacologic measures such as back strengthening ex-ercises to
counteract lumbar lordosis, avoidance of improper postural and
movement habits (for example, with Alexander technique), heat and
cold packs, and massages are the first line of management. If these
are not effective, a slow and step-wise approach to systemic
analgesics is needed. However, since this is a manifestation of
increased renal mass and is not attribut-able to individual cysts,
cyst decortication procedure has limit-ed to no role in management
of this type of pain.
2) Pain directly related to enlarged renal cysts: Individ-ual
expanding cysts can directly cause pain. This can be due to
stretching of the renal capsule, traction on the renal ped-icle or
compression of nearby structures. Pain severity does not always
correlate with cyst size; relatively small cysts can be a source of
significant pain and sometimes very large cysts produce no or
minimal discomfort. This pain is often localized to the anterior
abdomen, and less frequently to the back. Since a single or few
underlying cysts are responsible, pain is well lo-
calized and patients are often able to provide a finger-point
lo-calization of the epicenter of pain. Focal tenderness is usually
appreciated in the same area. These cysts may be amenable to
drainage, which can effectively control pain while avoiding the
side effects of systemic analgesics (in particular, the detrimental
effects of non-steroidal anti-inflammatory drugs on renal func-tion
and central nervous system effects of narcotic analgesics).
Cyst drainage can be achieved by any of the following
approach-es: needle aspiration, open surgical
decortication/marsupial-ization or laparoscopic cyst decortication
(LCD). Percutaneous aspiration is the least invasive. However,
fluid frequently re-ac-cumulates leading to recurrence of symptoms
and hence, it is not recommended for management of cyst-related
pain [5]. In the marsupialization/decortication procedure, a slit
is made in the wall of the cyst and the wall is excised or the
edges of the slit are sutured to the external surface; the site
remains open and drains freely. As a result, fluid is not allowed
to re-accumu-late and symptoms are less likely to recur. Surgical
marsupial-ization of renal cysts for pain management was first
described over a century ago [6]. Several case series documented
efficacy of this procedure in achieving pain control [7,8].
However, re-ports of deterioration of renal function after
undergoing this procedure led to a diminished enthusiasm for this
treatment modality for many decades [9,10]. In particular, Bricker
and Patton published in the New England Journal of Medicine in 1957
a report of two ADPKD cases whose creatinine clearance (Cr Cl)
values decreased immediately after surgery for cyst decompression,
and remained reduced at one year follow up [9]. Based on these
observations and recent evidence at that time that cystic nephrons
in ADPKD retain functional activity [11], they concluded that
destruction of a large number of af-fected nephrons in a polycystic
kidney (even with diminished reserve) will further compromise its
functional ability, there-by discrediting the procedure as
potentially detrimental. The procedure was resurrected in the 1980s
when He et al and Ye et al documented that this procedure could be
used safely and effectively for pain control in ADPKD, without any
acceleration of decline in renal function [12,13]. The use of a
laparoscopic approach for cyst decortication for cyst-related pain
in ADPKD patients was first reported by Teichman and Hulbert in
1995 in a report of six patients all of whom had failed to achieve
pain control with percutaneous aspiration [14,15]. Advantages of
using a laparoscopic approach over open surgery include less
post-operative pain, shorter hospital stay, quicker recovery and
improved cosmetic outcomes with equivalent pain relief. As such,
once the decision to pursue treatment with invasive measures is
made, LCD is the preferred technique. Contempo-rary laparoscopic
cyst decortication is accomplished by dis-secting larger cysts away
from surrounding peri-renal fat. The cyst wall is then excised at
its intersection with normal renal parenchyma and the internal cyst
wall is inspected and cauter-ized to avoid re-accumulation of
fluid, marsupialization with sutures is now considered
unnecessary.
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Jacobs Publishers 3
Cite this article: Garg N. Systematic Employment of Laparoscopic
Decortication for Maximal Pain Control in Polycystic Kidney
Disease. J J Nephro Urol. 2014, 1(2): 11.
Our guidelines for proceeding with LCD, which differ from prior
reports, are noted below and address which cysts and how many cysts
should be targeted. The role of underlying renal insufficiency
should factor into this decision making. In the literature, the
usual approach has been to decorticate as many of the larger cysts
as possible and to puncture and drain as many of the smaller cysts
as possible. Short term and long term limitations of this approach
are noteworthy: first, pain relief is maintained at 2 years
duration in only a little over 50% of the patients. In one
prospective study of 30 ADPKD patients undergoing open cyst
reduction surgery, only 62% were pain free at 2 year follow up
[16]. More recently, similar results have been reported with LCD
[17-19]. We postulate that lack of sustained relief from pain
observed in a large percentage of the patients undergoing LCD may
be, at least in part, due to sub-optimal implementation of this
technique:
a) Inappropriate selection of candidates: When the pain is
related to the mechanical effects of an overall increase in to-tal
kidney volume (as opposed to individual cysts), these pro-cedures
are unlikely to yield significant benefit.
b) Poor localization of culprit cysts responsible for
symptoms.
Secondly, anywhere from 200 to 600 cysts are usually treated at
each time. This blind and extensive decortication and drain-age
increases the total operative time as well as the likelihood for
intra-operative and post-operative complications [17,18]. Dunn et
al reported an analysis of 15 patients with ADPKD who underwent a
total of 21 LCDs; mean number of cysts marsupialized per procedure
was 204 (range 11 to 635) and mean operative time was 5.5 hours
(range 4.5 to 6.6 hours). At a mean follow up of 2.2 years, 11
patients (73%) reported an average of 62% improvement in pain, with
only transient or no improvement/ worsening in the other 4 (27%).
Average inpatient time after the procedure was 3.2 days. Three
patients in this series developed encapsulated collections of
extravasat-ed urine (also known as urinomas) postoperatively,
warrant-ing more procedures. In another series by Lee et al of 35
LCD procedures in 29 ADPKD patients, every detectable cyst within 2
mm of the renal surface was treated [18]. Mean operating time was
4.9 hours (range 2.6 to 6.6 hours); average number of cysts treated
per patient was 220 cysts (range 4 to 692). More than 50%
improvement in pain was noted in only 52% of the treated patients
at 24 months follow up. Urinomas developed in three patients
necessitating ureteral stent placement in all of them. Whether
there is any benefit from such extensive de-cortication,
particularly at the expense of these complications has not been
evaluated.
Thirdly, data regarding impact of LCD on natural progression of
renal insufficiency are mixed. Many uncontrolled case series
document that Cr Cl is unchanged after LCD [15,16,20-22]. Oth-
er studies have shown a vulnerability to renal function decline,
particularly if there is baseline impairment of kidney function
[17,19,23]. In the study of 29 ADPKD patients mentioned ear-lier,
all five patients with a preoperative Cr Cl of less than 30 ml/min
experienced worsening of renal function post opera-tively, and one
patient with normal renal function at baseline had a 22% drop in Cr
Cl at 24 months follow up [18]. Whether LCD itself alters the
progression of renal insufficiency remains an open question.
However, if the decline in renal function is a consequence of the
procedure itself, it is conceivable that doing more targeted and
less extensive decortications will improve renal outcomes. This is
especially relevant if renal in-sufficiency is already present,
where all renal compensatory mechanisms are already being fully
employed and there is lit-tle remaining reserve.
Methods
We describe a systematic approach to using LCD for manage-ment
of cyst-related pain in ADPKD, and illustrate its success-ful use
in 3 patients who underwent a total of 4 procedures. The steps of
this approach are outlined below:
1) To be eligible for this procedure, careful history and
phys-ical examination are required to establish whether the pain is
related to individual cysts and is severe and long-standing enough
to warrant invasive therapy:
i) The ADPKD patient must be able to provide a single finger
pinpoint location of reproducible chronic pain, usually in the
anterior abdomen, for at least 6 months in duration.
ii) The area of maximal tenderness is at the pinpoint
location.
iii) Pain must be moderate to severe and impacts the daily
quality of life. A visual analog scale (VAS) of pain is more than
5/10 (averaging 7/10 in our cohort).
iv) Maximal conservative therapy regimens have failed, but not
including chronic narcotic use. Patients ability to tolerate
various analgesics should factor into decision mak-ing.
2) The area of maximal pain/pinpoint tenderness is identified
using a skin marker in conjunction with ultrasonog-raphy (USG).The
aim of the radiologic evaluation is communi-cated to the
radiologist prior to the procedure.
3) Abdominal USG is used to identify the culprit cyst(s) that
are directly beneath the marked skin area and measure at least 4 cm
in the biggest dimension. In certain instances, ad-ditional
computed tomography/ magnetic resonance imaging (MRI) may be
required for better delineation prior to proceed-
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Jacobs Publishers 4
Cite this article: Garg N. Systematic Employment of Laparoscopic
Decortication for Maximal Pain Control in Polycystic Kidney
Disease. J J Nephro Urol. 2014, 1(2): 11.
ing with LCD.4) Following this, an experienced urologist who is
famil-iar with the nuances of surgical management in ADPKD
pa-tients employs the LCD technique, never unroofing more than
three cysts at a time.
Results
Case 1:
45 year old male with ADPKD and stage IV chronic kidney dis-ease
(CKD) with estimated glomerular filtration rate (eGFR) of 27
ml/min/m2 presented with localized right upper quad-rant pain. The
pain had been present for at least 3 years, and had been getting
worse over the preceding 6 months. It was described as constant and
non-radiating, and graded at 7 on 10-point VAS. Pain relief with
acetaminophen and tramadol was inadequate. Chronic use of
non-steroidal anti-inflamma-tory drugs was contraindicated due to
underlying renal insuf-ficiency. Given his teaching
responsibilities, he was unable to tolerate narcotic analgesics
during the daytime due to their sedative effects. Physical
examination was notable for bilater-ally palpable kidneys; with
severe tenderness on light anteri-or palpation in the right upper
quadrant. Using USG, a 5.7 cm was identified anteriorly in the
right kidney directly beneath the area of maximum discomfort
(Figure 1a). A second large cyst, 6.5 cm in the biggest dimension,
was identified inferior-ly (Figure 1b). Only these two cysts were
decorticated using the LCD technique (as demonstrated in video 1).
Jackson-Pratt (JP) drain was removed on post-operative day (POD) 1
and he was discharged to home on POD 2. The procedure resulted in
complete relief of pain on the right side that was maintained at
his last follow up at 34 months. The repeated and sustained VAS at
each visit was 1-2/10, down from 7/10.
He presented a year and a half after the initial procedure with
focal pain in left upper abdominal quadrant that was charac-terized
as constant, non-radiating and 8/10 in severity on the VAS. The
marked skin area of tenderness corresponded to the site of pain.
USG identified one cyst 4.7 cm in largest dimen-sion directly below
this area. A JP drain was not required and patient as discharged to
home on POD 2. Again, LCD directed at this single cyst resulted in
complete resolution of pain; pa-tient remained pain free (VAS 1/10)
at his last follow up at 15 months.
No differences were seen between pre-operative and
post-op-erative serum creatinine values on either of two instances.
However, he has had progressive CKD consistent with the nat-ural
course history of ADPKD, with the most recent eGFR 12 ml/min/1.73m2
and is undergoing evaluation for renal trans-plantation.
Figure 1a. A 5.7 cm x 5.4 cm cyst is identified in the anterior
aspect of the right kidney of patient in Case 1 directly beneath
the area of maximum pain/ tenderness.
Figure 1b. Another 6.5 x 5.1 cm cyst is identified in the right
kidney inferior to the cyst depicted in Figure 1a.
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Jacobs Publishers 5
Cite this article: Garg N. Systematic Employment of Laparoscopic
Decortication for Maximal Pain Control in Polycystic Kidney
Disease. J J Nephro Urol. 2014, 1(2): 11.
Case 2:
51 year old male with left-sided dominant ADPKD and normal renal
function with serum creatinine of 1 mg/dL presented with worsening
pain and discomfort on the left side of two years duration. Pain
severity was rated at 5/10-point VAS; this was associated with a
sensation of constant abdominal fullness and increased abdominal
girth. Bending forward and sleeping on his left side worsened this
discomfort. A large (20.8 cm x 16.2 cm) cyst originating from the
lower pole of the left kidney was visualized on USG; this was
significantly enlarged com-pared with his MRI from three years ago
when he had no pain (Figure 2). This large cyst was excised
(drained a total of 1.7 L of straw-colored fluid). Additionally,
two adjacent large cysts were also unroofed. He tolerated the
procedure well, was dis-charged to home the day after surgery and
remained symptom free at his last follow up 10 months after the
procedure (re-peated VAS 1/10). Patients renal function was not
affected; se-rum creatinine 10 months after the procedure was 0.9
mg/dL.
Case 3:
39 year old with ADPKD and preserved renal function with serum
creatinine of 0.9 mg/dL first presented to us for evalu-ation of
left flank pain of at least two to three years duration. This pain
was present all the time, being 7/10-point VAS and requiring near
maximal doses of tramadol on a daily basis. The pain episodically
radiated down his back to his left lower ex-tremity. Tenderness on
deep palpation was appreciated in the area where the patient
reported maximal pain. Corresponding to this area, two adjacent
cysts were identified in the lower pole of the left kidney on USG
(4.4 cm and 3.8 cm in largest dimension). These two cysts were
unroofed. Patient required placement of a JP drain that was removed
on POD1, following
which the patient was discharged. He reported complete
res-olution of his symptoms (VAS 1/10), which was maintained at
follow up six months after the procedure. This has been his longest
pain free interval in over 3 years. Additionally, his se-rum
creatinine remained unchanged.
Additional information regarding total operative time, length of
hospitalization and complications is outlined below:
Discussion
Pain is a common symptom in ADPKD, often occurring early in the
course of the disease and frequently leading to diagno-sis.
However, it is vastly under-recognized and its management remains
suboptimal; these results in unnecessary suffering, decreased
quality of life and lost productivity [24]. Acute pain can occur as
a result of cyst rupture and hemorrhage, pyelo-nephritis, cyst
infection (known as pyocystis) or nephrolithi-asis. As outlined
earlier, chronic pain in ADPKD can be a con-sequence of either the
effects of increased total kidney volume on the spinal column and
paraspinal muscles, or stretching of the capsule, compression or
pull created by the expanding cysts (resulting in irritation of
nerves in the renal capsule, re-nal parenchyma or intrarenal
collecting system). As with all other types of chronic pain,
comprehensive, step-wise and personalized approach involving
non-pharmacologic, pharma-cologic and surgical approaches is key to
achieving effective relief from pain [4]. This article focuses on
the appropriate and specific circumstance use of a LCD procedure to
offer long term relief/cure from pain while avoiding the side
effects of systemic medications.
The previous reported strategy with decortication procedures in
management of cyst-related pain in ADPKD has been to drain as many
cysts close to the surface of the kidney as possible. Not only has
this proven ineffective in providing long term relief in a large
percentage of the cases, it is associated with prolonged operative
and hospitalization times, and major post-operative complications
(eg., urinary leaks leading to formation of urino-mas). Most
concerning is the potentially detrimental effect on renal function
associated with unfocused broad scale unroof-
Figure 2. Directly underneath the area of maximum discomfort, a
large 20.8 cm x 16.2 cm cyst is identified in the
lower pole of the left kidney of patient in Case 2.
Additional information regarding total operative time, length of
hospitalization and complications is outlined below:
Case Number of cysts decorticated/ unroofed
Duration of procedure
Duration of hospitalization (from procedure to discharge)
Complications
Follow up
1 (Right) 2 63 minutes 2 days None No pain at 37 months
1 (Left) 1 64 minutes 2 days None No pain at 18 months
2 3 98 minutes 1 day None No pain at 13 months
3 2 64 minutes 1 day None No pain at 8 months
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Jacobs Publishers 6
Cite this article: Garg N. Systematic Employment of Laparoscopic
Decortication for Maximal Pain Control in Polycystic Kidney
Disease. J J Nephro Urol. 2014, 1(2): 11.
ing of cysts, especially in the face of pre-existing renal
insuf-ficiency where compensatory mechanisms have already been
fully exhausted. A more focused and limited approach has not been
reported, and no guiding principles are available regard-ing which
and how many cysts can be safely decorticated for pain control in
patients with ADPKD. To our knowledge, this case series represents
the first report documenting systematic employment of LCD with
specific criteria for targeting culprit cysts. Careful history and
physical examination combined with protocol-driven use of imaging
are outlined to identify patients whose chronic pain is
attributable to enlarging individual cysts and likely to benefit
from LCD. In three patients, who under-went a total of four
procedures, sustained complete resolution of pain was observed.
Operative time was dramatically lower than as reported in previous
studies. Blood loss was minimal. No major or minor complications
were observed and patients were discharged to home within 1 to 2
days. While the patient in case 1 has had progressive chronic
kidney disease over the last three years, it is notable that his
renal function parameters immediately after the surgery were
unchanged compared to pre-procedure. His projected slope of decline
in renal function was consistent with the natural course history of
his ADPKD and unaffected by surgery. Patients in cases 2 and 3 had
nor-mal renal function at baseline, and their serum creatinine
val-ues remained unchanged at last follow up.
In summary, this case series highlights that LCD is a useful
tool in the armamentarium for management of chronic pain in
management. Focused LCD aiming cysts responsible for patient
symptoms identified through thorough clinical and radiographic
evaluation can increase the success rate of the procedure while
minimizing adverse effects. The importance is noted of a
multi-disciplinary approach involving nephrolo-gy, urology and
radiology, all committed to pain relief in those with ADPKD.
Further long term studies in a large cohort are required to
establish the effectiveness of this approach.
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