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A Patient With Loin Hematuria Syndrome and Chronic Flank Pain Treated With Pulsed Radiofrequency of The Splanchnic Nerves

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CASE REPORT

A Patient With Loin Hematuria Syndrome and Chronic Flank


Pain Treated With Pulsed Radiofrequency of the
Splanchnic Nerves
Susan M. Moeschler, MD, Bryan C. Hoelzer, MD, and Jason S. Eldrige, MD

the onset of acute right-sided flank pain accompanied by hema-


Introduction: Chronic abdominal and flank pain can be multi- turia. Subsequent medical workup revealed a right-sided renal vein
factorial and difficult to treat. Loin pain hematuria syndrome thrombosis. He was consequently treated with warfarin, and fol-
(LPHS) is a rare clinical cause of chronic abdominal and flank pain low-up computed tomography imaging at 2 months demonstrated
and is a diagnosis of exclusion with limited treatment options, complete thrombosis resolution. However, the patient continued to
ranging from medications to renal autotransplantation or even report severe right flank pain that evolved into bilateral flank pain.
nephrectomy in resistant cases. The patient also had proteinuria and lower extremity edema and
was diagnosed with membranous glomerulonephritis after renal
Case Description: A 50-year-old man with a history of recurrent biopsy. His edema and proteinuria improved with rituximab, but
nephrolithiasis secondary to hypercalcemia presented to the pain the disabling bilateral flank pain persisted.
clinic with bilateral flank pain. After failed conservative medical Potential causes for the patient’s symptoms included neph-
management, the decision was made to proceed to interventional rolithiasis, tumors, and the more rare “nut-cracker syndrome”—
modalities. He responded for a short duration to a splanchnic nerve which refers to the compression of a renal vein between the aorta
block and subsequently had a longer analgesic response to pulsed and the superior mesenteric artery7—that were ruled out by
radiofrequency (PRF) ablation to the splanchnic nerves. imaging and other diagnostic evaluations. He was ultimately
Discussion: LPHS is a difficult clinical scenario to diagnose and diagnosed with LPHS, as his pain was persistent for >6 months,
treat. Conservative options are often unsuccessful, but the more was consistent with the distribution of his flank pain, and had not
extreme interventions such as renal autotransplantation and improved with resolution of the clot. It is notable that he retained
nephrectomies are invasive and not always effective. In this case normal kidney functions.8
report, we describe the novel use of PRF to the splanchnic nerves as The patient had a difficult time identifying any exacerbating
an alternative treatment modality for patients with LPHS. or alleviating factors for his pain, even with the use of a detailed
Although the exact mechanism of action of PRF on nerve tissue is pain journal. He described the pain as a dull ache in his bilateral
unclear, its indication in pain management requires further flanks with intermittent sharp exacerbations. His visual analog
research and discussion. Our patient experienced substantial and scale pain scores ranged from 5 to 10 on a 11-point scale. Physical
sustained relief of his flank pain. PRF may be a viable option for examination findings were unremarkable. Drug therapies, includ-
patients with LPHS. ing amitriptyline, gabapentin, and pregabalin, were minimally
effective and were eventually discontinued because of side effects
Key Words: pulsed radiofrequency, flank pain, loin pain hematuria and lack of analgesic benefit. Acupuncture was also tried without
syndrome any discernible benefit. He was then initiated on oxycodone sus-
tained release, 10 mg every 12 hours, in addition to hydro-
(Clin J Pain 2013;29:e26–e29) morphone, 2 mg every 4 hours as needed for pain.
Upon initial evaluation at the pain clinic, it was decided to
proceed with bilateral splanchnic nerve blocks for diagnostic and
hronic abdominal and flank pain can be multifactorial1
C and difficult to treat. Loin pain hematuria syndrome
(LPHS) is a rare clinical cause of chronic abdominal and
therapeutic benefit. He underwent the first bilateral splanchnic
nerve blocks with 10 mL of 0.50% bupivacaine and triamcinolone
20 mg and reported a >50% reduction of his bilateral flank pain
flank pain and is a diagnosis of exclusion.2,3 Some of these for approximately 6 weeks. However, the analgesic duration waned
with 2 subsequent blocks. Thus, it was decided to proceed with
patients have comorbid psychiatric issues, including pulsed radiofrequency (PRF) ablation to the bilateral splanchnic
somatoform disorders, and may undergo procedural nerves, specifically at the T12 and L1 vertebral levels.
interventions,4,5 such as renal denervation, kidney auto-
transplantation, or nephrectomy, all in an attempt to provide
symptomatic relief of pain.6 SURGICAL TECHNIQUE
The classic retrocrural approach was performed at the
T12 and L1 vertebral bodies (Fig. 1). These vertebral bodies
CASE REPORT were identified using an anterior-posterior view with fluo-
A 50-year-old man with multiple comorbidities was referred
roscopic imaging. Entry points were marked using an
to the pain clinic at the request of his attending nephrologist. The
patient had initially presented to the emergency department with
oblique entry point just under the rib at the T12 level and
just under the transverse process at the L1 level. An insu-
lated 20-G, 5-inch 10-mm active curved tip needle (Baylis
Received for publication August 29, 2012; revised December 19, 2012;
accepted February 10, 2013.
Medical Company, QC, Canada) was introduced using
From the Department of Anesthesiology and Pain Medicine, Mayo intermittent fluoroscopic guidance until the needle was
Clinic, Rochester, MN. adjacent to the vertebral body. Lateral fluoroscopic views
The authors declare no conflict of interest. were thereafter obtained to be certain that the needle tip
Reprints: Bryan C. Hoelzer, MD, Department of Anesthesiology and
Pain Medicine, Mayo Clinic, 200 1st Street SW, Rochester, MN
was located at the junction between the anterior third and
55905 (e-mail: hoelzer.bryan@mayo.edu). the middle third of the vertebral body. This was repeated at
Copyright r 2013 by Lippincott Williams & Wilkins the L1 level bilaterally to complete the placement of 4

e26 | www.clinicalpain.com Clin J Pain  Volume 29, Number 11, November 2013
Clin J Pain  Volume 29, Number 11, November 2013 Pulsed Radiofrequency of the Splanchnic Nerves

FIGURE 1. Splanchnic nerve anatomy, lateral and axial views. The gray shading depicts the anticipated retrocrural location of the
splanchnic nerves, as they traverse the posterior mediastinum around the T12-L1 vertebral levels. Note that the celiac plexus itself is
anterolateral to the aorta, at the level of the celiac artery.

needles (Figs. 2, 3). After negative aspiration, 0.25 mL of treatment is difficult as evidenced by the various treatments
Omnipaque was injected under live fluoroscopy and did not described in the literature including intraureteral cap-
show any evidence of intradiscal or intravascular spread. saicin,10 hypnotherapy,11 and intrathecal opioids.12 Hebert
Sensory testing was thereafter performed at a 50 Hz et al13 described the use of angiotensin-converting enzyme
frequency using escalating voltage. At 2 V, the patient com- inhibitors in the management of LPHS to decrease glo-
plained of the onset of severe abdominal and flank pain merular hemorrhage and therefore pain. There have been
consistent with his typical chronic pain. The PRF treatment reports of surgical denervation,14 neurostimulation of the
was then initiated at a frequency of 2 Hz and a pulse width of splanchnic nerves,15 and renal autotransplantation, in
20 ms for a total duration of 120 seconds for each needle which a kidney and the proximal portion of the ureter is
(Pain Management RF Generator; Baylis Medical Company, completely excised from the patient and then reimplanted
QC, Canada). The maximum temperature of each needle was into the iliac fossa, thus preserving renal function.16,17
recorded and was noted to never exceed 421C throughout the Autotransplantation is not a guaranteed success in regard
procedure. This PRF procedure was repeated 3 times for each to permanent analgesia.16
needle position, for a total of 6 minutes of treatment at each The splanchnic nerves (greater, lesser, and least) are
site. Each needle was then injected with 10 mg of tri- composed of the preganglionic nerves originating from the
amcinolone mixed with 0.25% bupivacaine for a total volume
of 1.5 mL per level. The patient had immediate relief of his
pain and the acute sharp exacerbations became less frequent
and less severe. His pain significantly improved such that he
was rarely using any opioid medication. At his 6-month
follow-up in the pain clinic, the patient subjectively reported a
95% improvement of his right flank pain. Unfortunately, the
patient’s left-sided pain began to return to baseline intensity
at 2 months. However, the patient reported improvement in
his overall pain and his functional capacity, including being
able to participate more fully in activities of daily living. The
patient also reported a decrease in his use of oral opioid
medications from daily use to only 3 to 4 times per week.
Although the patient’s left side flank pain only responded for
a short duration, he was satisfied with his global improve-
ment and did not wish to undergo further treatments.

DISCUSSION
LPHS is a clinical diagnosis of exclusion in patients
who experience hematuria and flank and loin pain without
loss of renal function or pathologic explantation for the
former. This clinical conundrum is challenging in its
pathophysiology, diagnosis, and treatment. The exact cause
remains elusive but it has been suggested that the hematuria
may be attributable to a thin glomerular basement mem- FIGURE 2. Anteroposterior view of needles at the T12 and L1
brane, which creates an occlusion within the renal tubules vertebral levels for pulsed radiofrequency of the splanchnic
and thus flank pain because of capsular expansion.9 The nerves.

r 2013 Lippincott Williams & Wilkins www.clinicalpain.com | e27


Moeschler et al Clin J Pain  Volume 29, Number 11, November 2013

treated with PRF.23 In their report, the splanchnic nerves


were treated bilaterally at the T12 level. In our case, we
elected to treat bilaterally at the T12 and L1 levels with the
hope that the additional treatment sites would result in
greater clinical effects and longer duration of relief. Fur-
thermore, in the aforementioned case series, interventions
aimed at the splanchnic nerves have been used for the
treatment of pancreatic-derived pain in contrast to this
case, which is attributed to a renal origin.
The PRF treatments in this patient resulted in near
complete resolution of his right-sided pain at 6 months and
substantial shorter term benefit on his left-sided pain. It is
unclear why the patient’s left-sided flank pain was less
responsive. One possible explanation is that the active
portions of the left-sided needle tips were not in as close
proximity to the left-sided splanchnic nerves when com-
pared with the right side. Ideally, the procedure would have
been repeated on the left side with slightly different needle
location with the hope of getting a more sustained result.
However, the patient was satisfied with his global
improvement and the benefits included pain intensity
reduction and decreased frequency of paroxysmal flank
pain attacks. This therapeutic response is similar to patients
FIGURE 3. Lateral view of needle placement at T12 and L1 for studied with chronic pancreatitis who underwent RFA to
pulsed radiofrequency of the splanchnic nerves. the splanchnics; 40% of patients had a 50% reduction in
pain with a decrease in opiate consumption.19 Our results
were also similar to patients with chronic pancreatitis who
intermediolateral cell column of T5-T12, as well as visceral underwent treatment with PRF to the splanchnic nerves.23
afferents from the abdomen, that carry nociceptive informa-
tion from the viscera. Anatomically, this splanchnic inner-
vation is bounded by the vertebral body medially, the pleura CONCLUSIONS
laterally, the posterior mediastinum ventrally, and the pleural Patients with LPHS are difficult to manage, in part
attachment to the vertebra dorsally.18 The reliable anatomic because of a general lack of objective diagnostic findings
location of these structures, in combination with their to explain their severe pain. Once the diagnosis is made,
accessibility from a posterior approach, makes splanchnic the pain can be more aggressively treated, although this
nerve block an attractive alternative to anterior celiac plexus often requires a multidisciplinary approach, which in-
blockade in patients with chronic abdominal pain. cludes psychology/psychiatry, medications, and procedural
Splanchnic nerve blocks and radiofrequency lesioning interventions.
have been performed in the past for patients with chronic We recognize that this is a case report describing PRF
pancreatitis pain. Raj et al19 have previously described of the splanchnic nerves for severe pain associated with
a radiofrequency technique to the splanchnic nerves for this LPHS. However, this treatment strategy should be con-
indication. This technical report was followed by a positive sidered for other abdominal pathology and associated pain
study by Garcea et al,20 in which the authors reported the that is innervated by the splanchnic nerves. In regard to
effects of conventional radiofrequency ablation (RFA) on LPHS, it offers a less invasive treatment compared with
10 patients with chronic pancreatitis pain. Despite these more invasive surgical treatment options, namely, surgical
positive results, concerns over the long-term effects of denervation or renal autotransplantation. Given the pos-
conventional RFA of nerves remain. itive results reported by Brennan and the outcome of this
PRF ablation differs from conventional radio- patient, pursuit of a larger study of PRF treatment to the
frequency in that the temperatures do not increase above splanchnic nerves is of importance. We propose that PRF
421C and therefore, do not induce Wallerian degeneration of the splanchnic nerves be considered as an intermediate
or chromatolysis of the nerve-in-question. By utilizing PRF treatment and temporizing means for nonopioid pain con-
instead of a neurolytic agent, side effects such as inflam- trol for LPHS.
mation and necrosis are also avoided.4 The exact mecha-
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