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A rare case of non-insulinoma pancreatic hypoglycaemia syndrome (niphs) in an adult due to localised islet cell hyperplasia-successfully managed by enucleation.
BMJ Case Rep 2011; 2011BC

Abstract

Persistent hyperinsulinemic hypoglycaemia is caused most commonly by an insulinoma in adults or by nesidioblastosis in neonates. In adults, localised islet cell hyperplasia is a rare disorder characterised by localised proliferation of islet cells. The authors present the case of a previously healthy non-obese middle aged female with new-onset severe hypoglycaemia. Laboratory findings confirmed a case of hyperinsulinemic hypoglycaemia. Endoscopic ultrasonography, intra-arterial calcium stimulation test and intraoperative ultrasonography showed a lesion in the uncinate process that was enucleated. Rest of the pancreas was normal. Histological examination and immunostaining of the resected tissue revealed pancreatic tissue with maintained acinar pattern consistent with diagnosis of localised islet cell hyperplasia. The patient did not have further episodes of hypoglycaemia following the procedure. Localised islet cell hyperplasia with such a very high insulin level is exceedingly rare in adult populations and not reported in literature. This diagnosis cannot be easily made through routine diagnostic laboratory or radiological procedures and likely requires a histological diagnosis. Management of this rare entity is by enucleation.

Authors+Show Affiliations

Department of Medicine, Maulana Azad Medical College, NewDelhi, India. drhemantnayak@gmail.comNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info available

Pub Type(s)

Case Reports
Journal Article

Language

eng

PubMed ID

22675010

Citation

Nayak, Hemanta K., et al. "A Rare Case of Non-insulinoma Pancreatic Hypoglycaemia Syndrome (niphs) in an Adult Due to Localised Islet Cell Hyperplasia-successfully Managed By Enucleation." BMJ Case Reports, vol. 2011, 2011.
Nayak HK, Sothwal A, Raizaida N, et al. A rare case of non-insulinoma pancreatic hypoglycaemia syndrome (niphs) in an adult due to localised islet cell hyperplasia-successfully managed by enucleation. BMJ Case Rep. 2011;2011.
Nayak, H. K., Sothwal, A., Raizaida, N., Daga, M. k., Agarwal, A. k., & Durga, G. (2011). A rare case of non-insulinoma pancreatic hypoglycaemia syndrome (niphs) in an adult due to localised islet cell hyperplasia-successfully managed by enucleation. BMJ Case Reports, 2011, doi:10.1136/bcr.07.2011.4554.
Nayak HK, et al. A Rare Case of Non-insulinoma Pancreatic Hypoglycaemia Syndrome (niphs) in an Adult Due to Localised Islet Cell Hyperplasia-successfully Managed By Enucleation. BMJ Case Rep. 2011 Oct 11;2011 PubMed PMID: 22675010.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - A rare case of non-insulinoma pancreatic hypoglycaemia syndrome (niphs) in an adult due to localised islet cell hyperplasia-successfully managed by enucleation. AU - Nayak,Hemanta K, AU - Sothwal,Arpit, AU - Raizaida,Nishant, AU - Daga,Mradul kumar, AU - Agarwal,Anil kumar, AU - Durga,Garima, Y1 - 2011/10/11/ PY - 2012/6/8/entrez PY - 2011/1/1/pubmed PY - 2013/11/6/medline JF - BMJ case reports JO - BMJ Case Rep VL - 2011 N2 - Persistent hyperinsulinemic hypoglycaemia is caused most commonly by an insulinoma in adults or by nesidioblastosis in neonates. In adults, localised islet cell hyperplasia is a rare disorder characterised by localised proliferation of islet cells. The authors present the case of a previously healthy non-obese middle aged female with new-onset severe hypoglycaemia. Laboratory findings confirmed a case of hyperinsulinemic hypoglycaemia. Endoscopic ultrasonography, intra-arterial calcium stimulation test and intraoperative ultrasonography showed a lesion in the uncinate process that was enucleated. Rest of the pancreas was normal. Histological examination and immunostaining of the resected tissue revealed pancreatic tissue with maintained acinar pattern consistent with diagnosis of localised islet cell hyperplasia. The patient did not have further episodes of hypoglycaemia following the procedure. Localised islet cell hyperplasia with such a very high insulin level is exceedingly rare in adult populations and not reported in literature. This diagnosis cannot be easily made through routine diagnostic laboratory or radiological procedures and likely requires a histological diagnosis. Management of this rare entity is by enucleation. SN - 1757-790X UR - https://www.unboundmedicine.com/medline/citation/22675010/A_rare_case_of_non_insulinoma_pancreatic_hypoglycaemia_syndrome__niphs__in_an_adult_due_to_localised_islet_cell_hyperplasia_successfully_managed_by_enucleation_ L2 - http://casereports.bmj.com/cgi/pmidlookup?view=long&pmid=22675010 DB - PRIME DP - Unbound Medicine ER -