Lesson of the month 2: Houssay phenomenon – hypopitutarism leading to remission of diabetes

Pramanik, Subhodip; Bhattacharjee, Rana; Mukhopadhyay, Pradip; Ghosh, Sujoy

doi:10.7861/clinmedicine.16-3-294

Cited by 5 publications

(5 citation statements)

References 9 publications

(11 reference statements)

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“…Several studies showed that panhypopituitarism due to pituitary hemorrhage/infarction (including Sheehan's syndrome) induced a remarkable reduction of insulin doses in patients with type 1 diabetes [3,4,13,14]. A complete amelioration of diabetes induced by adrenal insufficiency was reported in patients with type 2 diabetes [5], but it has never been reported in a patient with type 1 diabetes with decreased insulin secretory capacity to an insulindependent level as observed in our patient.…”

Section: Discussioncontrasting

confidence: 51%

“…Glucocorticoids increase blood glucose levels by increasing hepatic gluconeogenesis and inhibiting the uptake and utilization of glucose in peripheral tissues. Glucocorticoid deficiency can lead to an amelioration of glycemic control and/or a reduction of the insulin dose in patients with diabetes [2][3][4][5], but it is unclear whether the hyperglycemia in type 1 diabetes could be ameliorated without insulin treatment when complicated with adrenal insufficiency. Here, we describe the case of a patient who latently developed type 1 diabetes with endogenous insulin deficiency concurrently with adrenal insufficiency due to autoimmune hypophysitis.…”

Section: Adrenocorticotropic Hormone (Acth)mentioning

confidence: 99%

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Masked type 1 diabetes mellitus (T1DM) unveiled by glucocorticoid replacement: a case of simultaneous development of T1DM and hypophysitis in an elderly woman

et al. 2020

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As a rare condition characterized by inflammation of the pituitary gland, hypophysitis usually results in hypopituitarism and pituitary enlargement. The most critical outcome of hypopituitarism is caused by secondary adrenal insufficiency. Glucocorticoid deficiency is a life-threatening condition, and patients who develop this deficiency require prompt diagnosis and treatment. However, a delayed diagnosis of hypopituitarism may occur due to its non-specific clinical manifestations. A common presenting sign of glucocorticoid deficiency is hypoglycemia. The amelioration of hyperglycemia has been observed in diabetic patients with adrenal insufficiency. We report the case of a 70-year-old Japanese woman who had suffered from fatigue and anorexia for several months; she was admitted based on refractory hyponatremia (sodium 125-128 mEq/L) and hypoglycemia (glucose 58-75 mg/dL). Laboratory findings and magnetic resonance imaging findings led to the diagnosis of panhypopituitarism caused by autoimmune hypophysitis. After receiving 10 mg/day of hydrocortisone, the patient developed severe hyperglycemia (glucose >500 mg/dL). Undetectable C-peptide levels and positive results of both insulinoma-associated antigen-2 antibodies and insulin autoantibodies indicated that she had experienced a recent onset of type 1 diabetes. The pathophysiological process indicated that overt hyperglycemia could be masked by the deficient action of glucocorticoids even in a diabetic patient with endogenous insulin deficiency. This uncommon case reinforces the importance of the prompt diagnosis and treatment of hypopituitarism. Clinicians should remain aware of the possibility of hidden diabetes when treating hypoglycemia in patients with adrenal insufficiency.

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Section: Discussioncontrasting

confidence: 51%

Section: Adrenocorticotropic Hormone (Acth)mentioning

confidence: 99%

Masked type 1 diabetes mellitus (T1DM) unveiled by glucocorticoid replacement: a case of simultaneous development of T1DM and hypophysitis in an elderly woman

et al. 2020

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“…Several studies showed that panhypopituitarism induced a remarkable reduction in insulin doses in patients with T1DM [ 22 , 23 ]. A complete amelioration of diabetes induced by adrenal insufficiency was reported in patients with type 2 diabetes [ 24 ], but it has never been reported in a patient with T1DM with decreased insulin secretory capacity to an insulin-dependent level as observed in our patient.…”

Section: Discussioncontrasting

confidence: 50%

Nivolumab-induced hypophysitis followed by acute-onset type 1 diabetes with renal cell carcinoma: a case report

et al. 2021

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Background Immune checkpoint inhibitors have recently become widely used for the management of advanced cancer patients. During the development of immune checkpoint inhibitors (ICPIs), it was quickly recognized that they are associated with autoimmune or autoinflammatory side effects. These toxicities are known as immune-related adverse events (irAEs): common endocrine irAEs include hypophysitis and thyroid dysfunction, and uncommon irAEs include type 1 diabetes mellitus (T1DM). Case presentation A 62-year-old Japanese man with metastatic renal cell carcinoma was treated with sunitinib followed by the 10th cycle of treatment with the ICPI nivolumab. He had already had thyroiditis and hypophysitis due to these anti-cancer drugs. On admission, he showed an extremely elevated plasma glucose level (601 mg/dl) and a low C-peptide level, and was diagnosed with acute T1DM. The patient was treated with intravenous fluid infusion and continuous insulin infusion. On the second day, he was switched to multiple daily injections of insulin therapy. Since these treatments, his blood glucose levels have been stable and he has been treated with an additional 10 ICPI treatments for renal cell carcinoma for over a year. Conclusions Treatment with ICPIs is expected to increase in the future. There may be cases in which their use for cancer treatment is inevitable despite the side effects. As long as treatment with ICPI continues, multiple side effects can be expected in some cases. It is important to carefully observe the side effects that occur during ICPI treatment and to provide appropriate treatment for each side effect.

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“…Hypopituitarism could lead to recurrent hypoglycemia in diabetic patients and to a reduction in their insulin requirements as a result of adrenocorticotropic hormone deficiency (the Houssay phenomenon). [3,4] Obscure recurrent hypoglycemia requires endocrinologic tests to clarify possible underlying hypocortisolism. [5]…”

Section: Discussionmentioning

confidence: 99%

Severe hypoglycemia and hyponatremia caused by hypopituitarism in a female patient with type 1 diabetes

Zheng

Qiu²,

H³

2019

Medicine

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Rationale: The misdiagnosis of hypopituitarism is common due to its rarity and its nonspecific clinical manifestations. Our case report highlights the importance of critical evaluation regarding hypopituitarism as a cause of recurrent hypoglycemia, hyponatremia, and gastrointestinal symptoms in patients with T1DM, as misdiagnosis might be fatal to the patient. Patient concerns: We herein report the case of 35-year-old female patient who presented with 18 years of history of well-controlled type 1 diabetes mellitus and a 6-month history of recurrent nausea and vomiting, generalized weakness, hyponatremia, and severe hypoglycemia, despite a reduction in the dose of insulin. She was considered as having “type 1 diabetes and gastroparesis.” Four months later, she was diagnosed with hypothyroidism, and 25 μg/d of levothyroxine was prescribed. However, the levothyroxine had to be discontinued 1 week later because of frequent vomiting by the patient. Diagnosis: Further evaluation in our hospital revealed low-normal adrenocorticotropic hormone, low-normal serum cortisol, and low 24-hours urinary cortisol excretion. Secondary hypothyroidism and hypogonadotropic hypogonadism were also demonstrated. Based on the endocrinological findings, she was diagnosed with hypopituitarism possibly due to lymphocytic hypophysitis. Diabetic nephropathy was another diagnosis made after kidney biopsy. Interventions: The patient was treated with 100 mg/d of hydrocortisone intravenously for 2 weeks. After that, she continued on 15 mg/d of prednisone, and then 25 μg/d of levothyroxine was administered. Outcomes: The patient's insulin requirement increased to a premorbid level, the severe hypoglycemia resolved, the physical discomforts were alleviated, and blood electrolytes returned to normal. Lessons: This uncommon case reinforced the significance of a timely diagnosis and appropriate treatment of hypopituitarism. We recommend that physicians focus their awareness on this potentially life-threatening disease, as it is a condition potentially fatal to the patient if not recognized and treated.

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Lesson of the month 2: Houssay phenomenon – hypopitutarism leading to remission of diabetes

Cited by 5 publications

References 9 publications

Masked type 1 diabetes mellitus (T1DM) unveiled by glucocorticoid replacement: a case of simultaneous development of T1DM and hypophysitis in an elderly woman

Masked type 1 diabetes mellitus (T1DM) unveiled by glucocorticoid replacement: a case of simultaneous development of T1DM and hypophysitis in an elderly woman

Nivolumab-induced hypophysitis followed by acute-onset type 1 diabetes with renal cell carcinoma: a case report

Severe hypoglycemia and hyponatremia caused by hypopituitarism in a female patient with type 1 diabetes

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