Abstract

Objective. Noninsulinoma pancreatogenous hypoglycemia syndrome (NIPHS) is a disorder of endogenous hyperinsulinemia that is clinically distinguishable from insulinoma, with a greater preponderance after Roux-en-Y gastric bypass (RYBG). Hyperinsulinemic hypoglycemia can predispose to attenuation of counterregulatory hormone responses to hypoglycemia, and consequent suppression of the hypothalamic–pituitary–adrenal (HPA) axis. This case series describes 3 individuals who were diagnosed with adrenal insufficiency (AI) after undergoing RYGB, complicated by NIPHS. Methods. A retrospective chart review was performed for each individual. Chart review applied particular attention to the onset of hyperinsulinemic hypoglycemia following bariatric surgery and the dynamic testing leading to the diagnoses of NIPHS and AI. Results. In each case, reactive hypoglycemia ensued within months to years after RYGB. Cosyntropin stimulation testing confirmed the diagnosis of AI. Hydrocortisone therapy reduced the frequency and severity of hypoglycemia and was continued until successful medical and/or surgical management of hyperinsulinism occurred. Follow-up testing of the HPA axis demonstrated resolution of AI. In all cases, hydrocortisone therapy was finally discontinued without incident. Conclusion. We speculate that transient AI is a potential complication in patients who experience recurrent hyperinsulinemic hypoglycemia after RYGB. The putative mechanism for this observation may be attenuation of the HPA axis after prolonged exposure to severe, recurrent hypoglycemia. We conclude that biochemical screening for AI should be considered in individuals who develop post-RYGB hyperinsulinemic hypoglycemia. If AI is diagnosed, supportive treatment should be maintained until hyperinsulinemic hypoglycemia has been managed effectively.

Highlights

  • Disorders of endogenous hyperinsulinemia include insulinoma, persistent hyperinsulinemic hypoglycemia of infancy (PHHI), and noninsulinoma pancreatogenous hypoglycemia syndrome (NIPHS)

  • We report 3 individuals who underwent Roux-en-Y gastric bypass (RYGB) complicated by NIPHS and subsequently developed adrenal insufficiency (AI)

  • Two of the 3 patients with hyperinsulinemic hypoglycemia described in this report (Cases 1 and 2) developed postRYGB AI prior to medical or surgical therapy of hypoglycemia, and the third patient was diagnosed with post-RYGB AI after a distal pancreatectomy

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Summary

Introduction

Disorders of endogenous hyperinsulinemia include insulinoma, persistent hyperinsulinemic hypoglycemia of infancy (PHHI), and noninsulinoma pancreatogenous hypoglycemia syndrome (NIPHS). Infants with PHHI,[12,13] and in diabetic patients treated with excess exogenous insulin.[14,15] Insulinoma resection mitigates hyperinsulinemic hypoglycemic episodes and is associated with restoration of physiologic counterregulatory hormone response to hypoglycemia.[7,8,9,10,11] We report 3 individuals who underwent RYGB complicated by NIPHS and subsequently developed adrenal insufficiency (AI).

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