Browse

You are looking at 1 - 10 of 85 items

Takuya Higashitani Division of Endocrinology and Hypertension, Department of Cardiovascular and Internal Medicine, Graduate School of Medical Science, Kanazawa University, Kanazawa, Ishikawa, Japan

Search for other papers by Takuya Higashitani in
Google Scholar
PubMed
Close
,
Shigehiro Karashima Division of Endocrinology and Hypertension, Department of Cardiovascular and Internal Medicine, Graduate School of Medical Science, Kanazawa University, Kanazawa, Ishikawa, Japan

Search for other papers by Shigehiro Karashima in
Google Scholar
PubMed
Close
,
Daisuke Aono Division of Endocrinology and Hypertension, Department of Cardiovascular and Internal Medicine, Graduate School of Medical Science, Kanazawa University, Kanazawa, Ishikawa, Japan

Search for other papers by Daisuke Aono in
Google Scholar
PubMed
Close
,
Seigoh Konishi Division of Endocrinology and Hypertension, Department of Cardiovascular and Internal Medicine, Graduate School of Medical Science, Kanazawa University, Kanazawa, Ishikawa, Japan
Department of Internal Medicine, Keiju Medical Center, Nanao, Ishikawa, Japan

Search for other papers by Seigoh Konishi in
Google Scholar
PubMed
Close
,
Mitsuhiro Kometani Division of Endocrinology and Hypertension, Department of Cardiovascular and Internal Medicine, Graduate School of Medical Science, Kanazawa University, Kanazawa, Ishikawa, Japan

Search for other papers by Mitsuhiro Kometani in
Google Scholar
PubMed
Close
,
Rie Oka Division of Endocrinology and Hypertension, Department of Cardiovascular and Internal Medicine, Graduate School of Medical Science, Kanazawa University, Kanazawa, Ishikawa, Japan

Search for other papers by Rie Oka in
Google Scholar
PubMed
Close
,
Masashi Demura Department of Hygiene, Graduate School of Medical Science, Kanazawa University, Kanazawa, Ishikawa, Japan

Search for other papers by Masashi Demura in
Google Scholar
PubMed
Close
,
Kenji Furukawa Health Care Center, Japan Advanced Institute of Science and Technology, Nomi, Ishikawa, Japan

Search for other papers by Kenji Furukawa in
Google Scholar
PubMed
Close
,
Yuto Yamazaki Department of Pathology, Tohoku University Hospital, Sendai, Miyagi, Japan

Search for other papers by Yuto Yamazaki in
Google Scholar
PubMed
Close
,
Hironobu Sasano Department of Pathology, Tohoku University Hospital, Sendai, Miyagi, Japan

Search for other papers by Hironobu Sasano in
Google Scholar
PubMed
Close
,
Takashi Yoneda Division of Endocrinology and Hypertension, Department of Cardiovascular and Internal Medicine, Graduate School of Medical Science, Kanazawa University, Kanazawa, Ishikawa, Japan
Department of Health Promotion and Medicine of the Future, Kanazawa University, Kanazawa, Ishikawa, Japan

Search for other papers by Takashi Yoneda in
Google Scholar
PubMed
Close
, and
Yoshiyu Takeda Division of Endocrinology and Hypertension, Department of Cardiovascular and Internal Medicine, Graduate School of Medical Science, Kanazawa University, Kanazawa, Ishikawa, Japan

Search for other papers by Yoshiyu Takeda in
Google Scholar
PubMed
Close

Summary

Renovascular hypertension (RVHT) is an important and potentially treatable form of resistant hypertension. Hypercortisolemia could also cause hypertension and diabetes mellitus. We experienced a case wherein adrenalectomy markedly improved blood pressure and plasma glucose levels in a patient with RVHT and low-level autonomous cortisol secretion. A 62-year-old Japanese man had been treated for hypertension and diabetes mellitus for 10 years. He was hospitalized because of a disturbance in consciousness. His blood pressure (BP) was 236/118 mmHg, pulse rate was 132 beats/min, and plasma glucose level was 712 mg/dL. Abdominal CT scanning revealed the presence of bilateral adrenal masses and left atrophic kidney. Abdominal magnetic resonance angiography demonstrated marked stenosis of the left main renal artery. The patient was subsequently diagnosed with atherosclerotic RVHT with left renal artery stenosis. His left adrenal lobular mass was over 40 mm and it was clinically suspected the potential for cortisol overproduction. Therefore, laparoscopic left nephrectomy and adrenalectomy were simultaneously performed, resulting in improved BP and glucose levels. Pathological studies revealed the presence of multiple cortisol-producing adrenal nodules and aldosterone-producing cell clusters in the adjacent left adrenal cortex. In the present case, the activated renin-angiotensin-aldosterone system and cortisol overproduction resulted in severe hypertension, which was managed with simultaneous unilateral nephrectomy and adrenalectomy.

Learning points:

  • Concomitant activation of the renin-angiotensin-aldosterone system and cortisol overproduction may contribute to the development of severe hypertension and lead to lethal cardiovascular complications.

  • Treatment with simultaneous unilateral nephrectomy and adrenalectomy markedly improves BP and blood glucose levels.

  • CYP11B2 immunohistochemistry staining revealed the existence of aldosterone-producing cell clusters (APCCs) in the adjacent non-nodular adrenal gland, suggesting that APCCs may contribute to aldosterone overproduction in patients with RVHT.

Open access
Marina Yukina Department of Therapeutic Endocrinology, Endocrinology Research Centre (ERC), Moscow, Russia

Search for other papers by Marina Yukina in
Google Scholar
PubMed
Close
,
Nurana Nuralieva Department of Therapeutic Endocrinology, Endocrinology Research Centre (ERC), Moscow, Russia

Search for other papers by Nurana Nuralieva in
Google Scholar
PubMed
Close
,
Maksim Solovyev Department of Therapeutic Endocrinology, Endocrinology Research Centre (ERC), Moscow, Russia

Search for other papers by Maksim Solovyev in
Google Scholar
PubMed
Close
,
Ekaterina Troshina Department of Therapeutic Endocrinology, Endocrinology Research Centre (ERC), Moscow, Russia
Russian Academy of Sciences, Endocrinology Service, Department of Therapeutic Endocrinology, Endocrinology Research Centre (ERC), Moscow, Russia

Search for other papers by Ekaterina Troshina in
Google Scholar
PubMed
Close
, and
Evgeny Vasilyev Laboratory of Inherited Endocrine Disorders, Endocrinology Research Centre (ERC), Moscow, Russia

Search for other papers by Evgeny Vasilyev in
Google Scholar
PubMed
Close

Summary

Insulin autoimmune syndrome (Hirata’s disease) is a disorder caused by development of autoantibodies to insulin and manifested by hypoglycaemic syndrome. The overwhelming majority of physicians do not include it in the differential diagnosis of hypoglycaemic states because of a misconception of an extremely low prevalence of this condition. This results in unnecessary drug therapy and unjustified surgical interventions in patients that otherwise would be successfully treated conservatively. This disease is strongly associated with certain alleles of the HLA gene. In most cases, this condition develops in predisposed individuals taking drugs containing sulfhydryl groups. Formation of autoantibodies to insulin may be observed in patients with other autoimmune disorders, as well as in those with multiple myeloma or monoclonal gammopathy of undetermined significance. This paper presents the first Russian case report of insulin autoimmune syndrome in an adult patient.

Learning points:

  • Insulin autoimmune syndrome, Hirata’s disease, anti-insulin antibodies, and hypoglycaemia.

Open access
Kazuhisa Kusuki Department of Diabetes and Endocrinology, Kanto Central Hospital of the Mutual Aid Association of Public School Teachers, Setagaya-ku, Tokyo, Japan

Search for other papers by Kazuhisa Kusuki in
Google Scholar
PubMed
Close
,
Saya Suzuki Department of Diabetes and Endocrinology, Kanto Central Hospital of the Mutual Aid Association of Public School Teachers, Setagaya-ku, Tokyo, Japan

Search for other papers by Saya Suzuki in
Google Scholar
PubMed
Close
, and
Yuzo Mizuno Department of Diabetes and Endocrinology, Kanto Central Hospital of the Mutual Aid Association of Public School Teachers, Setagaya-ku, Tokyo, Japan

Search for other papers by Yuzo Mizuno in
Google Scholar
PubMed
Close

Summary

A 72-year-old man with no history of diabetes was referred to our department due to hyperglycemia during pembrolizumab treatment for non-small-cell lung carcinoma. His blood glucose level was 209 mg/dL, but he was not in a state of ketosis or ketoacidosis. Serum C-peptide levels persisted at first, but gradually decreased, and 18 days later, he was admitted to our hospital with diabetic ketoacidosis (DKA). The patient was diagnosed with fulminant type 1 diabetes (FT1D) induced by pembrolizumab. According to the literature, the insulin secretion capacity of a patient with type 1 diabetes (T1D) induced by anti-programmed cell death-1 (anti-PD-1) antibody is depleted in approximately 2 to 3 weeks, which is longer than that of typical FT1D. Patients with hyperglycemia and C-peptide persistence should be considered for hospitalization or frequent outpatient visits with insulin treatment because these could indicate the onset of life-threatening FT1D induced by anti-PD-1 antibodies. Based on the clinical course of this patient and the literature, we suggest monitoring anti-PD-1 antibody-related T1D.

Learning points:

  • Immune checkpoint inhibitors, such as anti-PD-1 antibodies, are increasingly used as anticancer drugs. Anti-PD-1 antibodies can cause immune-related adverse events, including T1D.

  • FT1D, a novel subtype of T1D, is characterized by the abrupt onset of hyperglycemia with ketoacidosis, a relatively low glycated hemoglobin level and depletion of C-peptide level at onset.

  • In patients being treated with anti-PD-1 antibody, hyperglycemia with C-peptide level persistence should be monitored through regular blood tests. Because of C-peptide persistence and mild hyperglycemia, it is possible to miss a diagnosis of life-threatening FT1D induced by anti-PD-1 antibody.

  • In particular, in patients who have no history of diabetes, hyperglycemia without DKA is likely to be the very beginning of anti-PD-1 antibody-induced T1D. Therefore, such patients must be considered for either hospitalization or frequent outpatient visits with insulin injections and self-monitoring of blood glucose.

Open access
Åke Sjöholm Division of Endocrinology and Diabetology, Department of Internal Medicine, Gävle Hospital, Gävle, Sweden

Search for other papers by Åke Sjöholm in
Google Scholar
PubMed
Close
,
Maria João Pereira Department of Medical Sciences, Uppsala University, Uppsala, Sweden

Search for other papers by Maria João Pereira in
Google Scholar
PubMed
Close
,
Thomas Nilsson Department of Medical Sciences, Uppsala University, Uppsala, Sweden

Search for other papers by Thomas Nilsson in
Google Scholar
PubMed
Close
,
Torbjörn Linde Department of Medical Sciences, Uppsala University, Uppsala, Sweden

Search for other papers by Torbjörn Linde in
Google Scholar
PubMed
Close
,
Petros Katsogiannos Department of Medical Sciences, Uppsala University, Uppsala, Sweden

Search for other papers by Petros Katsogiannos in
Google Scholar
PubMed
Close
,
Jan Saaf Department of Internal Medicine, Västmanland Hospital Köping, Köping, Sweden

Search for other papers by Jan Saaf in
Google Scholar
PubMed
Close
, and
Jan W Eriksson Department of Medical Sciences, Uppsala University, Uppsala, Sweden

Search for other papers by Jan W Eriksson in
Google Scholar
PubMed
Close

Summary

Type B insulin resistance syndrome (TBIRS) is a very rare autoimmune disorder with polyclonal autoantibodies against the insulin receptor, resulting in severe and refractory hyperglycemia. Described here is a patient who within a few months after the onset of autoimmune type 1 diabetes increased her insulin requirements more than 20-fold; despite this she had considerable difficulty maintaining a plasma glucose value of <40–60 mmol/L (720–1100 mg/dL). On suspicion of TBIRS, the patient was started on tapering dose of glucocorticoids to overcome the autoimmune insulin receptor blockade, resulting in an immediate and pronounced effect. Within days, insulin requirements decreased by 80–90% and plasma glucose stabilized around 7–8 mmol/L (126–144 mg/dL). The presence of antibodies to the insulin receptor was detected by immunoprecipitation and binding assays. After a 4-month remission on low maintenance dose prednisolone, the patient relapsed, which required repeated plasmaphereses and immune column treatments with temporarily remarkable effect. Mixed and transient results were seen with rituximab, mycophenolic acid and bortezomib, but the glycemic status remained suboptimal. Lack of compliance and recurrent infections may have contributed to this.

Learning points:

  • Type B insulin resistance syndrome (TBIRS) is a very rare autoimmune disorder with acquired polyclonal autoantibodies against the insulin receptor, resulting in severe and refractory hyperglycemia.

  • We describe here a young patient in whom, a few months after the onset of a regular autoimmune diabetes, insulin requirements in a short time increased more than 20-fold, but despite this, the plasma glucose level could be kept at <40–60 mmol/L only with considerable difficulty. Did this patient have TBIRS?

  • On suspicion of TBIRS, the patient was started on tapering glucocorticoids to overcome the autoimmune insulin receptor blockade, resulting in an immediate and pronounced effect; within days insulin requirements decreased by 80–90% and plasma glucose stabilized around 7–8 mmol/L.

  • The presence of antibodies to the insulin receptor was detected by immunoprecipitation and binding assays.

    After a 4-month remission on low maintenance dose prednisolone, the patient relapsed, which required repeated plasmaphereses with temporarily remarkable effect.

  • TBIRS should be considered in diabetic patients whose glycemia and/or insulin requirements are inexplicably and dramatically increased.