Surgery for Obesity and Related Diseases 9 (2013) e25– e27
Case report
Laparoscopic sleeve gastrectomy in Chinese female patient with Prader–Willi syndrome and diabetes Haoyong Yu, M.D.a,1, Jianzhong Di, M.D.b,1, Weiping Jia, Ph.D.a,* a
Department of Endocrinology and Metabolism, Shanghai Jiao Tong University Affiliated Sixth People’s Hospital, Shanghai Diabetes Institute, Shanghai Clinical Center of Diabetes, Shanghai Key Laboratory of Diabetes Mellitus, Shanghai, China b Department of General Surgery, Shanghai Jiao Tong University Affiliated Sixth People’s Hospital, Shanghai, China Received July 19, 2012; accepted July 25, 2012
Keywords:
Laparoscopic sleeve gastrectomy; Prader–Willi syndrome; Morbid obesity; Diabetes mellitus
Prader–Willi syndrome (PWS) is a complex genetic disorder localized to chromosome 15 and is considered the most common genetic cause of life-threatening obesity. The cause of death in most subjects with PWS has been related to complications of massive obesity. Conventional treatments with behavioral and dietary approaches under the guidance of an expert dietitian have proved unsuccessful. Thus, bariatric surgery remains the last choice for morbidly obese children with PWS. However, the appropriate surgical technique remains a matter of discussion [1]. Laparoscopic sleeve gastrectomy (LSG) is known as a beneficial weight loss procedure for high-risk patients [2]. We report a Chinese female patient with PWS and associated morbid obesity and type 2 diabetes mellitus (T2DM) who underwent LSG, followed by substantial weight loss and T2DM remission. Case report In April 2011, a 17-year-old female patient, who had been diagnosed with PWS according to the consensus diagnostic criteria [3], was admitted to our hospital for surgical treatment of obesity and T2DM. She had experienced neonatal hypotonia with poor sucking and feeding difficulties that gradually improved with age. During her childhood, she manifested im1
These two authors contributed equally to this work. *Correspondence: Weiping Jia, Ph.D., Department of Endocrinology and Metabolism, Shanghai Jiao Tong University Affiliated Sixth People’s Hospital, Shanghai Diabetes Institute, Shanghai Clinical Center of Diabetes, Shanghai 200233, China. E-mail:
[email protected]
paired satiety, growth failure, and excessive weight gain. Thus, she had marked progressive respiratory difficulties and severe daytime sleepiness. Behavioral approaches combined with food restriction were ineffective. In August 2010, physical examination found that her fasting plasma glucose was 11.3 mmol/L, postprandial plasma glucose was 15.6 mmol/L, glycosylated hemoglobin was 8.0%. With a diagnosis of severe
Table 1 Clinical parameters of patient before and after LSG Parameter
Weight (kg) BMI (kg/m2) Hemoglobin (g/dL) Total cholesterol (mmol/L) Total triglycerides (mmol/L) HDL cholesterol (mmol/L) LDL cholesterol (mmol/L) FPG (mmol/L) 2hPG (mmol/L) HbA1c (%) Insulin (U/mL) HOMA-IR SFA (cm2) VFA (cm2) EWL (%)
Before
After
April 2011
June 2011
September 2011
July 2012
94.2 46.7 140 6.55 1.65 1.22 4.61 6.1 8.1 6.9 27.2 7.37 593.1 118.8 —
85.5 43.3 130 6.86 2.05 .85 5.09 5.8 7.2 — 20.1 5.18 — — 15.2
79.0 39.2 131 5.99 1.60 1.09 3.91 4.3 4.6 5.6 8.3 1.58 534.7 64.2 26.6
68.0 33.7 124 4.62 1.08 1.00 2.87 4.1 4.8 4.9 4.02 .65 426.5 46.9 45.8
BMI ⫽ body mass index; HDL ⫽ high-density lipoprotein; LDL ⫽ low-density lipoprotein; FPG ⫽ fasting plasma glucose; 2hPG ⫽ 2-h plasma glucose; HbA1c ⫽ glycated hemoglobin A1c; HOMA-IR ⫽ Homeostasis Model Assessment of Insulin Resistance; SFA ⫽ subcutaneous fat area; VFA ⫽ visceral fat area; EWL ⫽ excess weight loss.
1550-7289/13/$ – see front matter © 2013 American Society for Metabolic and Bariatric Surgery. All rights reserved. http://dx.doi.org/10.1016/j.soard.2012.07.009
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H. Yu et al. / Surgery for Obesity and Related Diseases 9 (2013) e25– e27
Fig. 1. Abdominal magnetic resonance imaging scans showing reduction of subcutaneous fat area and visceral fat area. (A) Before surgery, (B) 5 months after surgery, (C) 15 months after surgery.
T2DM, she was treated with exenatide (5 g subcutaneous injection, twice daily). Before she underwent laparoscopic sleeve gastrectomy (LSG), she had lost almost 7 kg and her body mass index was 46.7 kg/m2. Furthermore, her fasting plasma glucose, postprandial plasma glucose, and glycosylated hemoglobin had all improved. On May 5, 2011, she underwent LSG without operative complications. The gastric sleeve had an estimated retained volume of 50 cm3. Postoperatively, she was given iron, calcium, vitamin D, minerals, and multivitamins and guidelines for her diet to prevent nutritional deficiencies. She was reexamined (clinical examination
and blood tests) 2, 5, and 15 months after surgery. Her obesity and T2DM improved considerably after LSG, without the need for any antidiabetic medication. By July 2012 (15 mo postoperatively), she had complete remission of T2DM and marked improvement in insulin resistance and other clinical parameters without any postoperative complications. Laboratory tests showed no anemia or malnutrition (Table 1). Abdominal magnetic resonance imaging showed that the subcutaneous fat area had decreased somewhat and the visceral fat area was markedly reduced (Fig. 1). Her weight had also decreased, from 94.2 to 68 kg.
LSG and PWS / Surgery for Obesity and Related Diseases 9 (2013) e25– e27
Discussion Obesity is the main cause of morbidity and mortality in patients with PWS, and their life expectancy is directly related to the degree of obesity present. The genetically induced changes in their central nervous system, especially in the hypothalamus, with their probably altered perception of satiety might be the reason for the hyperphagia. Thus, nonoperative treatment has shown limited success in patients with PWS. If an operation is safe and effective and the obesity is clinically severe, a bariatric operation during childhood is justified [4]. Several surgical techniques have been reported for the treatment of morbid obesity in patients with PWS, such as truncal vagotomy, jejunoileal bypass, vertical banded gastroplasty [5], adjustable silicone gastric banding, and gastric bypass [6]. However, the optimal procedure is far from determined. In 2008, Scheimann et al. [7] reported that among those with PWS, 54% underwent biliopancreatic diversion, 29% gastric bypass, 18% placement of a bioenteric intragastric balloon, 5.4% jejunoileal bypass, 3.6% gastroplasty, 3.6% vertical banded gastroplasty, 1.8% silicone band gastroplasty, and 1.8% truncal vagotomy with division of the major nerve trunks [7]. Very few reports have been published of LSG in those with PWS. LSG is basically unknown as a standalone technique for bariatric surgery in children and adolescents. Recent data from adult studies have suggested that LSG might also be a safe, beneficial, and effective standalone approach [8]. Safe because LSG resulted in minor complications in ⬍5% and major complications requiring reoperation in ⬍2% of adult patients. The obvious advantages of LSG are effective weight loss without a foreign body placed and without a lifelong dissociation of the gastrointestinal tract. There was even some concern that PWS might be a contraindication for bariatric surgery because food obsession and mental retardation might lead to weight regain. Critics considered that LSG might result in long-term dilation of the remaining stomach sleeve. However, a 20-month follow-up study of 23 morbid obese patients has refuted this viewpoint [9]. Furthermore, Weiner et al. [10] agreed that the gastric sleeve should have a volume not much ⬎50 mL because weight loss could be insufficient and pouch dilation could occur. Two months after LSG, our patient’s T2DM had been relieved. The mechanism for T2DM remission after LSG is intriguing. Just as with other bariatric procedures, a rapid improvement in glucose handling occurred immediately after LSG. It has been postulated that a chronic increase in glucagon-like peptide-1 might result in an increase in -cell mass [11]. Many studies have shown that LSG can accelerate gastric emptying and increase the production of glucagon-like peptide-1 and peptide YY [12]. In addition, LSG might result in a significant reduction in fasting ghrelin levels. The markedly decreased ghrelin, in addition to increased peptide YY levels, after LSG are associated with the reduction in appetite and weight [13] and the improvement in glucose metabolism. After LSG, our patient also had a considerable decrease in the vis-
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ceral fat area compared with the subcutaneous fat area. It has been well documented that visceral fat is more strongly associated with metabolic risk factors than is subcutaneous fat [14]. Zhang et al. [15] found that bariatric surgery can reduced visceral adipose inflammation and improves endothelial function in those with T2DM. Although the follow-up period of our patient was not very long, the early results during the first 15 months have been more than satisfactory. In summary, LSG appears to be a safe and effective alternative for the surgical treatment of morbidly obese and patients with T2DM and PWS, because it offers good results in weight loss, T2DM remission, and a chance for prolonged survival. Disclosures The authors have no commercial associations that might be a conflict of interest in relation to this article. References [1] Sugerman HJ, Sugerman EL, DeMaria EJ, et al. Bariatric surgery for severely obese adolescents. J Gastrointest Surg 2003;7:102–7. [2] Hamoui N, Anthone GJ, Kaufman HS, Crookes PF. Sleeve gastrectomy in the high-risk patient. Obes Surg 2006;16:1445–9. [3] Holm VA, Cassidy SB, Butler MG, et al. Prader–Willi syndrome: consensus diagnostic criteria. Pediatrics 1993;9:398 – 402. [4] Anderson AE, Soper RT, Scott DH. Gastric bypass for morbid obesity in children and adolescents. J Pediatr Surg 1980;15:876 – 81. [5] Mason EE, Scott DH, Doherty C, et al. Vertical banded gastroplasty in the severely obese under age twenty-one. Obes Surg 1995;5:23–33. [6] Kobayashi J, Kodama M, Yamazaki K, et al. Gastric bypass in a Japanese man with Prader–Willi syndrome and morbid obesity. Obes Surg 2003;13:803–5. [7] Scheimann AO, Butler MG, Gourash L, et al. Critical analysis of bariatric procedures in Prader–Willi syndrome. J Pediatr Gastroenterol Nutr 2008;46:80 –3. [8] Himpens J, Dapri D, Cadière GB. A prospective randomized study between laparoscopic gastric banding and laparoscopic isolated sleeve gastrectomy: results after 1 and 3 years. Obes Surg 2006;16:1450 – 6. [9] Langer FB, Bohdjalian A, Felberbauer FX, et al. Does gastric dilatation limit the success of sleeve gastrectomy as a sole operation for morbid obesity? Obes Surg 2006;16:166 –71. [10] Weiner RA, Weiner S, Pomhoff I, Jacobi C, Makarewicz W, Weigand G. Laparoscopic sleeve gastrectomy—influence of sleeve size and resected gastric volume. Obes Surg 2007;17:1297–305. [11] Cummings DE. Gastric bypass and nesidioblastosis—too much of a good thing for islets? N Engl J Med 2005;353:300 –2. [12] Peterli R, Wòlnerhanssen B, Peters T, et al. Improvement in glucose metabolism after bariatric surgery: comparison of laparoscopic Rouxen-Y gastric bypass and laparoscopic sleeve gastrectomy: a prospective randomized trial. Ann Surg 2009;250:234 – 41. [13] Langer FB, Hoda R, Bohdjalian A, et al. Sleeve gastrectomy and gastric banding: effects on plasma ghrelin levels. Obes Surg 2005;15:1024 –9. [14] Taksali SE, Caprio S, Dziura J, et al. High visceral and low abdominal subcutaneous fat stores in the obese adolescent: a determinant of an adverse metabolic phenotype. Diabetes 2008;57:367–71. [15] Zhang H, Wang Y, Zhang J, Potter BJ, Sowers JR, Zhang C. Bariatric surgery reduces visceral adipose inflammation and improves endothelial function in type 2 diabetic mice. Arterioscler Thromb Vasc Biol 2011;31:2063–9.