Acute respiratory distress syndrome triggered by marked cytokine storm in a subject with diabetic ketoacidosis: A case report.

RATIONALE: Acute respiratory distress syndrome (ARDS) is an acute diffuse inflammatory lung injury. Many causes of acute direct and indirect lung injury have been described as possible initiators of ARDS. According to the literature data, ARDS could be a rare complication associated with the acute onset of diabetic ketoacidosis (DKA). Moreover, it has been suggested that cytokine release during DKA is involved in the above-mentioned acute clinical complications of DKA. PATIENTCONCERNS: A 48-year-old Japanese woman with a 4-year history of type 1 diabetes mellitus was brought to an emergency room with symptoms of deteriorated consciousness. Three days before, she was diagnosed with influenza A infection. DIAGNOSIS: Inflammation markers were markedly elevated and she was under DKA condition. Since her respiratory conditions were suddenly and markedly aggravated 2 days later, we diagnosed her as ARDS and continued systemic management with the ventilator.Interleukin-6 (IL-6) level was markedly elevated at the onset of ARDS, although IL-6 level was high at the onset of DKA. ARDS was suggested to be caused by marked cytokine storm and DKA. INTERVENTIONS: We continued to treat her hyperglycemic crises. Moreover, we continued systemic management with the ventilator. OUTCOMES: Approximately three weeks later, her general conditions were stabilized and ventilator management was stopped. We successfully treated her ARDS and hyperglycemic crises. LESSONS: This case is very important because it shows that DKA can induce cytokine storm, which leads to the onset of ARDS. Therefore, monitoring various cytokines such as IL-6, which are associated with ARDS during the period of treatment of DKA is beneficial.

Formation of large abscess tumor including gallstone in ileum during therapy for hyperosmolar hyperglycemic syndrome: a case report.

Hyperosmolar hyperglycemic status (HHS) is one of the most serious acute metabolic complications of diabetes mellitus (DM) and the patients with type 2 DM are immunocompromised host, especially when glycemic control is poor. Under hyperglycemic conditions, we have to pay much attention even for rare infection. Bouveret's syndrome is a rare type of gallstone ileus together with duodenal obstruction secondary to the passage of a stone through a cholecystoduodenal fistula. Herein, we reported a rare case with formation of large abscess tumor, including necrotic ileum and gallstone after taking therapy for HHS. She was under more rare conditions rather than Bouveret's syndrome, because large abscess tumor was formed not in the duodenal bulb but in the ileum together with necrotic ileitis and abdominal abscess with impacted gallstone. We should bear in mind that T2DM patients are immunocompromised host in particular under untreated or poorly controlled conditions and thereby they could have rare inflammatory diseases such as a large abscess tumor. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1007/s13340-021-00540-3.

Case Report: Markedly Long-Term Preservation of Pancreatic ß-Cell Function in a Subject With Elderly Onset of Type 1 Diabetes Mellitus Showing High-Titer Autoimmune Antibodies for Over 4 Years.

Type 1 diabetes mellitus (T1DM) is mainly triggered by autoimmune ß-cell destruction, usually leading to absolute insulin deficiency. Regarding the speed of ß-cell destruction, there are large variations depending on age. In some adult cases, sufficient ß-cell function is sometimes retained for a relatively long period and eventually they become dependent on insulin for survival. It is known that even in subjects with T1DM showing high titers of such antibodies, insulin secretory capacity is preserved under several conditions such as "honeymoon" period and slowly progressive T1DM (SPIDDM). Herein, we reported the acute onset T1DM subject with long-term preservation of ß-cell function, although his anti-GAD antibody and anti-IA-2 antibody titers were very high for more than 4 years. This case is very important in that his ß-cell function was preserved with dipeptidyl peptidase-4 inhibitor alone. This means that there are large variations in the speed of ß-cell destruction in the onset of T1DM.

Primary Epstein-Barr virus infection shortly after primary Cytomegalovirus infection: a case report.

BACKGROUND: Infectious mononucleosis (IM) and mononucleosis-like illnesses are common viral infectious diseases which are often accompanied by a high fever, pharyngitis and lymphadenopathy in adults, although such infection in childhood is generally subclinical. Most cases of IM are caused by the Epstein-Barr virus (EBV) or Cytomegalovirus (CMV). However, it is difficult to diagnose IM only with subjective symptoms, and thus EBV and CMV are nearly indistinguishable in clinical practice. CASE PRESENTATION: A 20-year-old healthy Japanese woman had a 2-day history of high fever and consulted us. She had sex for the first time 6 months earlier. Her virus antibodies showed that she was infected with primary CMV. About 5 months later, she again experienced high fever and lymph node enlargement at the posterior cervical region. Her virus antibodies showed that she was infected with primary EBV at that time. CONCLUSION: Herein, we report a healthy adult Japanese woman with primary EBV infection relatively soon after primary CMV infection. It is very interesting to compare the symptoms and/or clinical data after EBV and CMV infection in the same patient within a short period of time. Our patient was diagnosed based only on subjective symptoms, physical examination and laboratory data, without tests of such virus-related antibodies. Therefore, clinicians should bear in mind that primary EBV infection and/or primary CMV infection is possible when patients have symptoms such as high fever, pharyngitis and lymphadenopathy, even in healthy adults.

Alteration of ACTH and Cortisol Levels After Estradiol Valerate Treatment in a Male Subject With Gender Dysphoria: A Case Report.

Background: The number of subjects with gender dysphoria has been increasing. In general, male-to-female transsexual subjects are treated with estradiol valerate therapy. In this report, we showed the time course of ACTH and cortisol levels after estradiol valerate injection in a male subject with gender dysphoria. It seemed that alteration of estradiol levels influenced ACTH and cortisol levels via some pathway. Case presentation: A 31-year-old man with estradiol valerate therapy for gender dysphoria was referred due to an elevation of serum cortisol levels. She started hormone therapy at 26 years old. Her laboratory analyses showed an elevation of plasma ACTH and cortisol levels. There were no remarkable changes in the adrenal gland and pituitary gland. Her estradiol levels were elevated 7 days after estradiol valerate injection, but they were not detected 18 days after such treatment. Interestingly, plasma ACTH and serum cortisol levels were moderately decreased 7 days after estradiol valerate injection, but both were markedly elevated 18 days after such treatment. Conclusions: We should bear in mind the possibility of elevation in plasma ACTH and serum cortisol levels when we start estradiol valerate injection in subjects with gender dysphoria. In addition, we may need to check ACTH and cortisol levels when we use estrogen replacement therapy for a long period of time in subjects with gender dysphoria.

Severe hypertriglyceridemia in a subject with disturbed life style and poor glycemic control without recurrence of acute pancreatitis: a case report.

BACKGROUND: Hypertriglyceridemia is often observed as the result of lipid abnormality and frequently associated with other lipid and metabolic disorders. Aggravation of hypertriglyceridemia is caused by various conditions. However, severe hypertriglyceridemia is usually induced by an addition of some secondary clinical conditions such as uncontrolled type 2 diabetes mellitus (T2DM) and obesity with insulin resistance. CASE PRESENTATION: A 40-year-old man with 4-year history of dyslipidemia and T2DM visited after his interruption of therapy for about 1.5 years. His past history was acute pancreatitis. His life style was markedly disturbed, and he had a lot of risk factors for hypertriglyceridemia. Surprisingly, his serum triglyceride level was as high as 16,900 mg/dL. His aggravation and remission of hypertriglyceridemia were closely associated with the alteration of RLP-cholesterol levels in dyslipidemia and glycoalbumin and ketone body levels in T2DM. CONCLUSION: We report very severe hypertriglyceridemia, which seemed to be caused by markedly disturbed life style and poorly controlled T2DM. Total therapy with diet and drug for each disease is very important for the improvement of very severe hypertriglyceridemia. This case report suggests that very severe hypertriglyceridemia alone does not necessarily bring out acute pancreatitis, although it is very important to check pancreatitis markers in such a situation.

Acute exacerbation of chronic osteomyelitis triggered by aggravation of type 2 diabetes mellitus: a case report.

BACKGROUND: Osteomyelitis is an infection in a bone. Acute osteomyelitis is observed mainly in the long leg bones of children and is usually treated with antibiotics. On the other hand, in adults, subacute or chronic osteomyelitis is more common. Antibiotics therapy is not necessarily effective for chronic osteomyelitis, and sometimes a surgical operation is performed for its remission. Furthermore, in classification of osteomyelitis by cause, type 2 diabetes mellitus is one of most common conditions associated with osteomyelitis. It isCase presentation well known that a variety of complications are induced in patients with type 2 diabetes mellitus due to chronic hyperglycemia, inflammatory reaction, and immunodeficiency, especially when glycemic control is poor. CASE PRESENTATION: A 58-year-old Japanese man had acute exacerbation of chronic osteomyelitis triggered by aggravation of type 2 diabetes mellitus. He had acute osteomyelitis in his right lower leg in his babyhood. After this episode, he did not experience any pain in his leg for approximately 50 years; he felt acute pain in his right lower leg at the age of 50 when his glycemic control was very poor. He then started undergoing medical therapy for type 2 diabetes mellitus and, after an improvement in glycemic control, his pain was gradually mitigated. However, he did not take medicine for approximately 8 months at the age of 58. After the interruption, glycemic control became very poor and he felt the similar acute pain again in the same area. After improving glycemic control, his pain was gradually mitigated again as observed at the age of 50. CONCLUSIONS: Here we report a case of chronic osteomyelitis under poorly controlled diabetic conditions. Interestingly, chronic osteomyelitis was observed at the same position where acute osteomyelitis was observed in his babyhood. In addition, chronic osteomyelitis was repeatedly observed, and it seemed that its acute exacerbation was closely associated with aggravation of type 2 diabetes mellitus. We should bear in mind that type 2 diabetes mellitus is one of the major risk factors of osteomyelitis and that acute exacerbation of chronic osteomyelitis could be triggered by a disturbance of glycemic control in patients with type 2 diabetes mellitus.

Drug fever and acute inflammation from hypercytokinemia triggered by dipeptidyl peptidase-4 inhibitor vildagliptin.

A 69-year-old man started taking the dipeptidyl peptidase-4 inhibitor, vildagliptin. One week later, C-reactive protein and plasma immunoglobulin E levels were markedly elevated, and the vildagliptin was stopped. After the patient's laboratory findings were normalized, we decided to restart vildagliptin with the patient's agreement. The next day, he had a high fever, and C-reactive protein and procalcitonin levels were elevated. Although we failed to find a focus of infection, we started antibiotics therapy. Two days later, the high fever had improved, and the C-reactive protein level had decreased. A drug lymphocyte stimulation test showed a positive result for vildagliptin. We examined various kinds of cytokine and infection markers just before and after the treatment with vildagliptin. Finally, we diagnosed the patient with vildagliptin-induced drug fever, probably based on the increase of various inflammatory cytokine levels and the response to this. Taken together, we should be aware of the possibility of vildagliptin inducing drug fever and/or acute inflammation.

Hypoinsulinemic hypoglycemia triggered by liver injury in elderly subjects with low body weight: case reports.

Hypoglycemia is induced by many causes, especially over-dose of insulin or oral hypoglycemic agents in diabetic subjects. In such a case, hyperinsulinemic hypoglycemia is usually observed. On the other hand, it is important to classify secondary hypoglycemia and hypoinsulinemic hypoglycemia. Liver injury-induced hypoglycemia is one of the causes of hypoinsulinemic hypoglycemia but rarely observed in clinical practice. Herein, we experienced similar 2 cases of non-diabetic hypoinsulinemic hypoglycemia. Both of them were elderly subjects with low body weight. Furthermore, it is likely that hypoinsulinemic hypoglycemia in both subjects was triggered by severe liver injury, at least in part, due to possible limited liver glycogen store. In elderly subjects with low body weight and/or malnutrition, metabolism in the liver is reduced and glycogen accumulation is decreased. Such alteration brings out acute and marked liver injury, which finally leads to the onset of severe hypoglycemia. It is known that not only liver injury but also multiple organ failure could be induced due to extreme emaciation in subjects. It is likely that in elderly subjects with low body weight and/or malnutrition, multiple organ failure including liver failure could be induced due to the similar reason. Therefore, we should be very careful of such subjects in order to avoid the development of multiple organ failure which leads to life-threatening situations. In conclusion, we should keep in mind the possibility of hypoinsulinemic hypoglycemia when we examine severe liver injury, especially in elderly or starving subjects with low body weight and limited liver glycogen stores. LEARNING POINTS: It is important to classify secondary hypoglycemia and hypoinsulinemic hypoglycemia.Liver injury-induced hypoglycemia is one of the causes of hypoinsulinemic hypoglycemia but rarely observed in everyday clinical practice.Herein, we reported similar 2 cases of hypoinsulinemic hypoglycemia without diabetes presumably triggered by severe liver injury.In both cases, hypoglycemia was improved by glucose infusion, although their liver injury was not improved.We should keep in mind the possibility of hypoinsulinemic hypoglycemia when we examine severe liver injury, especially in elderly subjects with low body weight.

Clinical course of pituitary function and image in IgG4-related hypophysitis.

A 76-year-old man had a hypopituitarism including adrenal insufficiency, hypogonadism and hypothyroidism. Based on various findings including the swelling of the pituitary gland, increase of serum IgG4 level and abundant IgG4-positive plasma cell infiltration in immunostaining of the pituitary gland, we diagnosed this subject as IgG4-related hypophysitis. In general, a high-dose glucocorticoid treatment is effective for IgG4-related disease. His clinical symptom, laboratory data and adrenal insufficiency were almost improved without any therapy. The serum IgG4 level was decreased and pituitary size was normalized with hydrocortisone as physiological replacement. This case report provides the possibility that IgG4 level is decreased spontaneously or with physiological dose of glucocorticoid therapy. LEARNING POINTS: We performed the pituitary gland biopsy and histochemical examination glucocorticoid therapy in a subject with IgG4-related hypophysitis.This case report provides the possibility that IgG4 level is decreased spontaneously or with a physiological dose of glucocorticoid therapy. We reported the clinical course of IgG4-related hypophysitis without a high-dose glucocorticoid treatment, although there were a few reports about the retrospective examination.Although the patient had still higher IgG4 level compared to normal range, his clinical symptom disappeared and his laboratory data were improved.We should keep in mind the possibility of IgG4-related hypophysitis when we examine one of the uncertain causes of a hypopituitarism including adrenal insufficiency, hypogonadism and hypothyroidism.