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Year : 2021  |  Volume : 48  |  Issue : 2  |  Page : 107-109

Management of Gilbert's syndrome with low-dose, diluted hyperbaricity of spinal bupivacaine: A rare-rare combo

Department of Anaesthesia, Bangalore Baptist Hospital, Bengaluru, Karnataka, India

Date of Submission20-Jan-2021
Date of Acceptance06-May-2021
Date of Web Publication18-Aug-2021

Correspondence Address:
Reena Ravindra Kadni
Department of Anaesthesia, Bangalore Baptist Hospital, Bellary Road, Hebbal, Bengaluru - 560 024, Karnataka
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/jss.jss_6_21

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Gilbert's syndrome (GS) is an inherited unconjugated hyperbilirubinemia state. Regional anesthesia is recommended over general anesthesia in the management of these patients for surgeries. We describe anesthesia management of a young patient with GS for a below umbilical surgery, with modified spinal anesthesia by diluting the hyperbaricity and reducing the concentration in prone position. Selective sensory blockade with stable hemodynamics, faster recovery, and minimal stress response proved to be beneficial. When feasible, this modified spinal anesthesia can be opted for the management of short duration surgeries in patients with GS.

Keywords: Diluted hyperbaricity, Gilbert's syndrome, hemodynamics, prone position, spinal anesthesia

How to cite this article:
Kadni RR, Samuel LA, Archana S, Sravanthi A. Management of Gilbert's syndrome with low-dose, diluted hyperbaricity of spinal bupivacaine: A rare-rare combo. J Sci Soc 2021;48:107-9

How to cite this URL:
Kadni RR, Samuel LA, Archana S, Sravanthi A. Management of Gilbert's syndrome with low-dose, diluted hyperbaricity of spinal bupivacaine: A rare-rare combo. J Sci Soc [serial online] 2021 [cited 2021 Dec 6];48:107-9. Available from: https://www.jscisociety.com/text.asp?2021/48/2/107/324080

  Introduction Top

Gilbert's syndrome (GS) is a congenital nonhemolytic jaundice.[1] Its benign and chronic nature remains a concern for anesthetic management. Regional anesthesia is described safe for perineal surgery. We describe the use and effect of low-dose, diluted hyperbaricity of bupivacaine spinal anesthesia for perineal surgery. We present a patient with GS managed uneventfully with this scarcely practiced technique.

  Case Report Top

A 22-year-old male patient weighing 60 kg was planned for pilonidal sinus excision with flap repair. He was a known case of GS for 2 years. On examination, there was an icterus in the sclera; vitals and cardiovascular and respiratory system were normal. Investigations revealed: hemoglobin 15.6 g/dl, total count: 5200/mm3, platelets: 229 × 103, random blood sugar (RBS): 113 mg/dl, serum creatinine: 0.94, liver function tests (LFTs): Total protein: 8 g/dL, albumin: 4.8 g/dL, direct bilirubin: 0.64 mg/dL, total bilirubin: 4.81 mg/dL, alkaline phosphatase: 78 U/L, serum glutamic oxaloacetic transaminase (SGOT): 21 U/L, and serum glutamic pyruvic transaminase (SGPT): 17 U/L. Urine routine was normal. Coagulation profile was normal: Prothrombin time: 12.9/12 s, INR: 1.05, and APTT: 28.3/28 s. Written informed consent was taken from the patient.

Premedication was done with oral alprazolam 0.5 mg, the night before surgery. Intravenous (IV) fluid Ringer's lactate, 100 ml/h at 6 AM on the day of surgery was advised. Neuraxial anesthesia was planned.

In operation theater, hypoglycemia was identified with a fasting blood sugar of 60 mg%, with patient being asymptomatic. The patient was optimized with IV 25% dextrose 50 ml bolus. A prespinal anesthetic crystalloid preload with 500 ml Ringer's lactate solution was infused followed by a maintenance infusion of 10% dextrose solution. Baseline vitals were: pulse rate: 82/min and blood pressure (BP): 96/56 mmHg which improved to 116/64 mmHg. Repeat RBS after 15 min was 126 mg%.

The patient was explained about spinal anesthesia and the need for prone position for the surgery. Under aseptic precautions, spinal anesthesia was given with 26 G Quincke's needle (B Braun) in the left lateral position, and 5 ml of 0.1% diluted bupivacaine with 25 mcg fentanyl was given. The specific gravity of the solution was 1.015, tested in our hospital biochemical laboratory (Cobas u 411, Roche). The drug was prepared under sterile conditions by diluting 1 ml of 0.5% hyperbaric bupivacaine (Neon) with 3.5 ml of sterile distilled water and 0.5 ml of fentanyl. The patient was immediately positioned prone with a broad pillow support for upper shoulder and head, and 25°–30° low Trendelenburg position was given for a higher spread of the block [Figure 1]. After 15 min, sensory level to loss of cold sensation was T4 (result of high intrathecal injection volume), and motor Bromage scale of 3 with ability to flex feet. The patient was hemodynamically stable and comfortable throughout the procedure [Table 1]. Total duration of surgery was 85 min and supplemental analgesia was not required. Two-segment sensory regression was observed at 60 min. Total recovery from the motor was at 120 min, and the patient complained of mild pain at 240 min. Postoperative analgesia was managed with IV paracetamol 1 g Q8 h for 24 h.
Figure 1: Position of the patient in Trendlenberg postspinal anesthesia

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Table 1: Intraoperative haemodynamics and sensory-motor levels with diluted hyperbaricity spinal anaesthesia

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Postoperative LFT done showed a minor increase of total bilirubin. Total bilirubin: 5.06 mg%, direct bilirubin: 0.68 mg%, alkaline phosphatase: 73U, SGOT: 17U, and SGPT: 11U

The patient was discharged on the 2nd postoperative day without any concerns.

  Discussion Top

GS was first described by Augustine Gilbert and Pierre Lereboullet in 1901, which is the most common inherited cause of unconjugated hyperbilirubinemia. Incidence is 2%–13%.[2] The condition results from decreased function of the conjugating enzyme system bilirubin-uridine diphosphate glucuronyltransferase and decreased uptake of unconjugated bilirubin by hepatocytes.[3]

Factors known to aggravate the symptoms of GS are fasting, surgery, infection, exercise, fatigue, alcohol intake, and menstruation.[4] Anesthesia and surgery can be additional stress factors.

In the intraoperative period, various anesthetic considerations include planning, minimizing fasting period to prevent hypoglycemia and dehydration, maintenance of mean arterial pressure (MAP) within 10% of baseline or at least MAP >65 mmHg to ensure optimal hepatic perfusion, and avoidance of hypothermia and hypoglycemia. Polypharmacy, hepatotoxic drugs, and anesthetic drugs biotransformed by glucuronyltransferase in the liver should ideally be avoided. Anxiolysis and adequate analgesia to blunt neuroendocrine response need to be ensured. Target-controlled administration of drugs to avoid overdosing is required. Various surgical factors, such as reducing the duration of surgery, blood loss, tissue retraction during upper abdominal surgery, and excellent hemostasis, should also be taken into consideration.[5],[6]

GS can aggravate the risk of postoperative jaundice in absence of management of above factors.

Regional anesthesia is advised where appropriate and bupivacaine is the preferred agent.[6]

The baseline BP and blood sugar levels of the patient instigated us to plan for low-dose weak-concentration high-volume spinal anesthesia.

Excellent perioperative analgesia and hemodynamic stability were observed in patients undergoing anorectal surgery in jackknife position with 5 ml intrathecal hypobaric bupivacaine 0.1%.[7] Dramatic reductions in hypotension and use of vasopressors were observed when Ben-David et al. compared 4 mg intrathecal isobaric bupivacaine with 20 mcg fentanyl with higher doses in patients with hip fractures.[8]

Major effects on intrathecal spread depend on the densities of the cerebrospinal fluid and the drug solution used: Sinking effect of hyperbaric and floating effect of hypobaric and an interplay between density and patient position.[9]

In 1929, Gasser and Erlanger demonstrated that all local anesthetics (LAs) at lower concentrations block small fibers than larger fibers of the same type. As a group, unmyelinated fibers are resistant to LAs compared with larger myelinated A-δ fibers. Bupivacaine and ropivacaine are relatively selective for sensory fibers and can result in dominant sensory loss with lower concentrations of intrathecal drugs.[10]

Fentanyl is considered safe as it is metabolized in the liver by CYP3A4 primarily to norfentanyl (>99%).[6] We used fentanyl 25 mcg intrathecally as an additive which proved beneficial for postoperative analgesia. Paracetamol was used for 24 h and was not of much concern.

Diluted hyperbaricity was the choice to decrease the concentration of the drug, hence, to result in more sensory than motor block. This helped the patient to position himself in prone position. Hemodynamic stability was achieved with hydration, preloading, and Trendlenberg position. Minimal stress and faster recovery with low-dose, diluted hyperbaricity spinal bupivacaine with opioid additive can be a good option for infraumbilical procedures of short duration in patients with GS.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understand that name and initial will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship


Conflicts of interest

There are no conflicts of interest.

  References Top

Borlak J, Thum T, Landt O, Erb K, Hermann R. Moleculardiagnosis of a familial non hemolytic hyperbilirubinemia (Gilbert's syndrome) in healthy subjects. Hepatology 2000;32:792-5.  Back to cited text no. 1
Chapman RW, Collier JD, Hayes PC. Liver and Biliary Tract Disease. In: Boon NA, Colledge NR, Walker BR, editors. Davidson's Principles and Practice of Medicine. 20th ed. Philadelphia, USA: Churchill Livingstone Elsevier; 2008. p. 925-6.  Back to cited text no. 2
Ranjan RV, Ramachandran TR, Veliath DG, Coelho D. Perioperative management of a patient with Gilberts syndrome and rheumatic heart disease. Saudi J Anaesth 2012;6:289-91.  Back to cited text no. 3
Radu P, Atsmon J. Gilbert's syndrome–Clinical and pharmacological implications. Isr Med Assoc J 2001;3:593-8.  Back to cited text no. 4
Jena BR, Khandelwal A, Pandia MP, Singh GP. Anesthetic Management of a Patient with Gilbert's Syndrome for Spine Surgery: A case report. J Neuroanaesthesiol Crit Care 2020;07:107-9.  Back to cited text no. 5
Tzoneva D, Aleksiev E, Michova K, Stanimirov P. Perioperative management and anaesthetic considerations for adult patients with Gilbert's syndrome and oral cancer: Review and case report. Biotechnol Biotechnol Equip 2019;33:1182-86.  Back to cited text no. 6
Maroof M, Khan RM, Siddique M, Tariq M. Hypobaric spinal anaesthesia with bupivacaine (0.1%) gives selective sensory block for ano-rectal surgery. Can J Anaesth 1995;42:691-4.  Back to cited text no. 7
Ben-David B, Frankel R, Arzumanov T, Marchevsky Y, Volpin G. Minidose bupivacaine-fentanyl spinal anesthesia for surgical repair of hip fracture in the aged. Anesthesiology 2000;92:6-10.  Back to cited text no. 8
Hocking G, Wildsmith JA. Intrathecal drug spread. Br J Anaesth 2004;93:568-78.  Back to cited text no. 9
Butterworth JF. Clinical Pharmacology of Local Anesthetics. In: Cousins MJ, Carr DB, Horlocker TT, Bridenbaugh PO, editors. Cousins and Bridenbaugh's Neural Blockade: In Clinical Anesthesia and Pain Medicine. 4th ed. Philadelphia: Lippincott Williams and Wilkins; 2009. p. 96-113.  Back to cited text no. 10


  [Figure 1]

  [Table 1]


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