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2018 | 14 | 2 | 237–240
Article title

Diarrhoea in a 2-year-old boy – thinking beyond the gut

Content
Title variants
PL
Biegunka u 2-letniego chłopca – etiologia pozajelitowa
Languages of publication
EN
Abstracts
EN
We report a male child with an unusual, very rare cause of chronic diarrhoea – WDHA (watery diarrhoea, hypokalaemia, achlorhydria) syndrome. A 2-year-old boy presented with long-term diarrhoea, failure to thrive and a presumed diagnosis of coeliac disease. The failure of a gluten-free diet, permanent hypokalaemia and the progressive worsening of the patient’s condition led to the finding of an adrenal tumour (mature ganglioneuroma) with elevation of vasoactive intestinal polypeptide (VIP) – criteria for the diagnosis of WDHA syndrome. Complete symptoms resolution occurred after tumour excision.
PL
W niniejszej pracy opisujemy przypadek dziecka z przewlekłą biegunką o niezwykle rzadkiej etiologii, jaką jest zespół WDHA (watery diarrhoea, hypokalaemia, achlorhydria), w języku polskim częściej określany jako zespół Vernera–Morrisona. U dwuletniego chłopca występowały przewlekła biegunka oraz niedobór wzrostu i wagi, skutkując domniemanym rozpoznaniem celiakii. W wyniku dalszego postępowania diagnostycznego, wynikającego z braku efektów wprowadzonej diety bezglutenowej oraz utrzymującej się hipokaliemii przy pogarszającym się stanie pacjenta, ujawniono guza nadnerczy (dojrzały ganglioneuroma) oraz podwyższone stężenie VIP (vasoactive intestinal polypeptide, wazoaktywny peptyd jelitowy), które stanowią kryteria rozpoznania zespołu Vernera–Morrisona. Objawy ustąpiły całkowicie po chirurgicznym usunięciu guza.
Keywords
Discipline
Year
Volume
14
Issue
2
Pages
237–240
Physical description
Contributors
  • Department of Paediatrics, Western Ukrainian Specialized Children’s Medical Centre, Lviv, Ukraine
  • Department of Paediatrics, Western Ukrainian Specialized Children’s Medical Centre, Lviv, Ukraine
  • Department of Surgery, Western Ukrainian Specialized Children’s Medical Centre, Lviv, Ukraine
  • Department of Clinical Immunology and Allergology, Lviv National Medical University, Lviv, Ukraine
References
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  • 2. Verner JV, Morrison AB: Islet cell tumor and a syndrome of refractory watery diarrhea and hypokalemia.Am J Med 1958; 25: 374–380.
  • 3. Matsumoto KK, Peter JB, Schultze RG et al.: Watery diarrhea and hypokalemia associated with pancreatic islet cell adenoma. Gastroenterology 1966; 50: 231–242.
  • 4. Cruz-Escobar EDL, Juárez IG, Urbina S et al.: Verner–Morrison syndrome presenting as acute persistent diarrhea. J Clin Case Rep 2016; 6: 729.
  • 5. Dimitriadis GK, Weickert MO, Randeva HS et al.: Medical management of secretory syndromes related to gastroenteropancreatic neuroendocrine tumours. Endocr Relat Cancer 2016; 23: R423–R436.
  • 6. Parbhu SK, Adler DG: Pancreatic neuroendocrine tumors: contemporary diagnosis and management. Hosp Pract (1995) 2016; 44: 109–119.
  • 7. Kargl S, Tamesberger M, Pumberger W: Intractable diarrhea and a paravertebral mass. J Pediatr Gastroenterol Nutr 2013; 57: e11.
  • 8. Reindl T, Degenhardt P, Luck W et al.: [The VIP-secreting tumor as a differential diagnosis of protracted diarrhea in pediatrics]. Klin Padiatr 2004; 216: 264–269.
  • 9. Long RG, Bryant MG, Mitchell SJ et al.: Clinicopathological study of pancreatic and ganglioneuroblastoma tumours secreting vasoactive intestinal polypeptide (vipomas). Br Med J (Clin Res Ed) 1981; 282: 1767–1771.
  • 10. Adam O, Boia ES, Ilie R et al.: Ganglioneuroma. Jurnalul Pediatrului 2007; 10: 48–51.
  • 11. Lacayo NJ, Davis KL: Pediatric neuroblastoma. Available from: https://emedicine.medscape.com/article/988284-overview#a6 [cited: 9 October 2017].
  • 12. Kumar B, Upadhyaya VD, Rao RN et al.: Ganglioneuroblastoma as vasoactive intestinal polypeptide-secreting tumor: rare case report in a child. World J Endoc Surg 2015; 7: 47–50.
  • 13. Gilroy RK, Brand RE: WDHA syndrome clinical presentation. Available from: https://emedicine.medscape.com/article/183189- clinical [cited: 5 November 2017].
  • 14. Kibria R, Ahmed S, Ali SA et al.: Hypokalemic rhabdomyolysis due to watery diarrhea, hypokalemia, achlorhydria (WDHA) syndrome caused by vipoma. South Med J 2009; 102: 761–764.
  • 15. Singh HM, Teller T, Esrason KT et al.: VIPoma: a rare cause of acute diarrhea. Available from: http://www.hcplive.com/publications/surgical-rounds/2007/2007-09/2007-09_02 [cited: 14 January 2011].
  • 16. Gilroy RK, Brand RE: WDHA syndrome workup. Available from: https://emedicine.medscape.com/article/183189-workup [cited: 5 November 2017].
  • 17. Gilroy RK, Brand RE: WDHA syndrome treatment & management. Available from: https://emedicine.medscape.com/ article/183189-treatment [cited: 2 November 2017].
Document Type
article
Publication order reference
Identifiers
YADDA identifier
bwmeta1.element.psjd-604b750a-0c1c-48ce-91f4-d19be637dcab
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