CC BY 4.0 · European J Pediatr Surg Rep. 2022; 10(01): e9-e12
DOI: 10.1055/s-0042-1742780
Case Report

Indocyanine Green (ICG)-Guided Identification of Hypermetabolic Pancreatic Nodules in Focal Congenital Hyperinsulinism: A Case Report in a 3-Month-Old Infant

1   Department of Pediatric Surgery, La Paz Children's University Hospital, Madrid, Spain
,
1   Department of Pediatric Surgery, La Paz Children's University Hospital, Madrid, Spain
,
1   Department of Pediatric Surgery, La Paz Children's University Hospital, Madrid, Spain
,
María del Carmen Sarmiento
1   Department of Pediatric Surgery, La Paz Children's University Hospital, Madrid, Spain
,
Miriam Miguel-Ferrero
1   Department of Pediatric Surgery, La Paz Children's University Hospital, Madrid, Spain
,
Nuria Leal
1   Department of Pediatric Surgery, La Paz Children's University Hospital, Madrid, Spain
,
Saturnino Barrena
1   Department of Pediatric Surgery, La Paz Children's University Hospital, Madrid, Spain
,
Leopoldo Martínez
1   Department of Pediatric Surgery, La Paz Children's University Hospital, Madrid, Spain
2   Institute for Biomedical Research La Paz (IdiPaz), Network for Maternal and Children Health (SAMID), La Paz Children's Hospital, Madrid, Spain
› Institutsangaben
Funding This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

Abstract

Indocyanine green (ICG)-guided near-infrared fluorescence has been recently adopted in pediatric surgery, although its use in the treatment of congenital hyperinsulinism has not been reported. We present a case of focal congenital hyperinsulinism in which ICG-navigation with ICG was used during surgical treatment. A 3-month-old infant was referred to our institution from a peripheral hospital for episodes of persistent hypoglycemia since birth, with no response to intravenous treatment with diazoxide, octreotide, or hydrochlorothiazide. An abdominal positron emission tomography-computed tomography scan showed a hypermetabolic nodule in the proximal portion of the body of the pancreas, compatible with focal congenital hyperinsulinism. A heterozygous mutation in the ABCC gene (Ala1516Glyfs*19) frameshift type inherited from the father was identified, which supported this diagnosis. Laparoscopy-assisted surgery was performed with ICG-guided near-infrared fluorescence, with intravenous injection of 16 mg ICG (2 mg/mg), which allowed localization of the focal lesion in the body of the pancreas. The lesion was resected with bipolar electrocautery and intraoperative histological study confirmed complete resection. Plasma glucose values normalized 6 hours after surgery and the patient was discharged 5 days later. In conclusion, the use of ICG in the treatment of congenital hyperinsulinism helps to identify hypermetabolic pancreatic nodules, decreasing the likelihood of incomplete resection.



Publikationsverlauf

Eingereicht: 03. August 2021

Angenommen: 23. November 2021

Artikel online veröffentlicht:
08. Februar 2022

© 2022. The Author(s). This is an open access article published by Thieme under the terms of the Creative Commons Attribution License, permitting unrestricted use, distribution, and reproduction so long as the original work is properly cited. (https://creativecommons.org/licenses/by/4.0/)

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Rüdigerstraße 14, 70469 Stuttgart, Germany

 
  • References

  • 1 Alander JT, Kaartinen I, Laakso A. et al. A review of indocyanine green fluorescent imaging in surgery. Int J Biomed Imaging 2012; 2012: 940585
  • 2 Verbeek FP, Schaafsma BE, Tummers QR. et al. Optimization of near-infrared fluorescence cholangiography for open and laparoscopic surgery. Surg Endosc 2014; 28 (04) 1076-1082
  • 3 Nagaya T, Nakamura YA, Choyke PL, Kobayashi H. Fluorescence-Guided Surgery. Front Oncol 2017; 7: 314
  • 4 Lau CT, Au DM, Wong KKY. Application of indocyanine green in pediatric surgery. Pediatr Surg Int 2019; 35 (10) 1035-1041
  • 5 Esposito C, Del Conte F, Cerulo M. et al. Clinical application and technical standardization of indocyanine green (ICG) fluorescence imaging in pediatric minimally invasive surgery. Pediatr Surg Int 2019; 35 (10) 1043-1050
  • 6 Goldstein SD, Heaton TE, Bondoc A. et al. Evolving applications of fluorescence guided surgery in pediatric surgical oncology: a practical guide for surgeons. J Pediatr Surg 2021; 56 (02) 215-223
  • 7 Palladino AA, Stanley CA. A specialized team approach to diagnosis and medical versus surgical treatment of infants with congenital hyperinsulinism. Semin Pediatr Surg 2011; 20 (01) 32-37
  • 8 Adzick NS, De Leon DD, States LJ. et al. Surgical treatment of congenital hyperinsulinism: results from 500 pancreatectomies in neonates and children. J Pediatr Surg 2019; 54 (01) 27-32
  • 9 Verkarre V, Fournet JC, de Lonlay P. et al. Paternal mutation of the sulfonylurea receptor (SUR1) gene and maternal loss of 11p15 imprinted genes lead to persistent hyperinsulinism in focal adenomatous hyperplasia. J Clin Invest 1998; 102 (07) 1286-1291
  • 10 Lord K, Radcliffe J, Gallagher PR, Adzick NS, Stanley CA, De León DD. High risk of diabetes and neurobehavioral deficits in individuals with surgically treated hyperinsulinism. J Clin Endocrinol Metab 2015; 100 (11) 4133-4139
  • 11 Laje P, States LJ, Zhuang H. et al. Accuracy of PET/CT Scan in the diagnosis of the focal form of congenital hyperinsulinism. J Pediatr Surg 2013; 48 (02) 388-393
  • 12 Arya VB, Senniappan S, Demirbilek H. et al. Pancreatic endocrine and exocrine function in children following near-total pancreatectomy for diffuse congenital hyperinsulinism. PLoS One 2014; 9 (05) e98054
  • 13 Boni L, David G, Mangano A. et al. Clinical applications of indocyanine green (ICG) enhanced fluorescence in laparoscopic surgery. Surg Endosc 2015; 29 (07) 2046-2055