Ko Harada

Hyperthyroidism with Selective Immunoglobulin A Deficiency

A 38-year-old Japanese woman with a history of Graves’ disease (GD) presented with a chief complaint of fatigue. The results of examinations showed recurrent empyema, tonsillitis and respiratory infections. She had no family history of immunodeficiency. Her thyroid was slightly enlarged with increased blood flow by ultrasound (Picture A). Laboratory tests revealed selective IgA deficiency (SIgAD) with recurrent GD as follows: IgA, 2.6 mg/dL (normal: 110-410); IgG, 1,676 mg/dL (861-1747); IgM, 173.9 mg/dL (50-269); IgE, <10 IU/mL (<180); thyrotropin, 0.005 μIU/mL; free T3, 12.73 pg/mL; free T4, 3.99 ng/dL; and TSAb, 283% (<120). SIgAD was confirmed by immunofixation electrophoresis (Picture B). The prevalence of SIgAD in Japanese is very low, but patients with SIgAD are at an increased risk for the development of autoimmune diseases, including GD (1). It is possible that SIgAD and GD share predisposing genetic factors or that increased infection due to SIgAD precipitates autoimmune disorders, such as GD (2).

Female paraurethral leiomyoma successfully excised through a vaginal approach: A case report

Paraurethral leiomyomas are extremely rare, and few cases have been reported in the literature. Because of the rarity of the disease and its anatomical complexity, diagnosis and management is challenging for clinicians. Here, we herein report the case of a 50‐year‐old woman who complained of a protruding mass at the urethral opening. Magnetic resonance imaging revealed a sharply defined tumor measuring 43 × 36 × 26 mm in diameter, located in the anterior vaginal wall, compressing the urethra. Based on the perioperative diagnosis of leiomyoma, the paraurethral tumor was excised via the vaginal approach. The patient was discharged 4 days postoperatively without any complications. Pathological features confirmed the diagnosis of leiomyoma. This case implies that, although there is the possibility of complications due to the surgical procedure, tumor removal is a reasonable choice for management to exclude malignancy and relieve symptoms.

Giant adenomatous thyroid nodule compressing the trachea

This paper details the clinical history and management of a 20‐year‐old male patient who presented with a 2‐year history of a neck swelling which was gradually enlarging during the last 3 months. The tumor was histologically diagnosed as an adenomatous nodule of the thyroid gland.

Anterior spinal cord syndrome-“owl's eye sign”

Magnetic resonance imaging revealing abnormally high signal intensity in the thoracic cord, extending from T5 to T10 on a sagittal T2‐weighted image (Figure 1A). An axial T2‐weighted image showing “owls eye sign” involving central‐anterior cord (Figure 1B), which was consistent with anterior spinal cord syndrome.

Platypnea-orthodeoxia syndrome induced by multiple vertebral compression fractures and an atrial septal defect

Platypnea-orthodeoxia syndrome (POS) is a rare condition that is characterized by dyspnea and arterial oxygen desaturation, which worsen on standing and which are relieved by recumbency. We treated an 80-year-old woman with an atrial septal defect (ASD) who demonstrated POS following thoracic and lumbar vertebral compression fractures. The surgical closure of the ASD relieved her symptoms. The etiology might have been multiple compression fractures causing kyphosis and aortic distortion producing right atrial compression and increased right-to-left flow through the ASD. POS should be considered in the differential diagnosis of patients who develop dyspnea after vertebral compression fractures. The careful assessment of the patients history and clinical condition helps in the diagnosis of POS.

Multiple abscesses caused by repetitive intramuscular injections

A 52yearold man was referred to our hospital owing to fever of unknown origin lasting for 3 months. On examination, there was tenderness, swelling, and redness in both of his upper arms (Figure 1A). During a prior hospitalization, the patient had received as many as 250 intramuscular injections of hydroxyzine in his gluteus medius muscles and deltoid muscles bilaterally due to severe pain after an injury sustained in a traffic accident. Meropenem had been administered for 4 weeks in the previous hospital because of the fever of unknown origin. Blood cultures taken on the day of admission were negative. Computed tomography of his pelvis revealed bilateral cellulitis in his buttocks and abscess formation on his left side (Figure 2, arrows). Gdenhanced magnetic resonance imaging of his upper arms indicated the formation of a bilateral subcutaneous abscess 19 mm in diameter (Figure 3A,B, arrows). Antibiotics, including cefazolin and amoxicillin/ clavulanic acid, were administered for 6 weeks, and his symptoms improved (Figure 1B). Abscess formation must be recognized as a complication of inappropriate frequent intramuscular injections.1,2

A case of traumatic cardiopulmonary arrest with good neurological outcome predicted by amplitude-integrated electroencephalogram

Highlights • Traumatic cardiopulmonary arrest requires unique therapeutic approaches, although angiography and percutaneous coronary intervention are often required in other forms of arythmogenic cardiopulmonary arrest.• Amplitude-integrated electroencephalogram was useful for traumatic cardiopulmonary arrest to predict the neurological outcome in the early stage of clinical course.• Amplitude-integrated electroencephalogram may help clinicians to start invasive and intensive treatments in a case of traumatic cardiopulmonary arrest.

The clinical and hormonal characteristics of primary adrenal lymphomas: The necessity of early detection of adrenal insufficiency

Objective To analyze the clinical and endocrine characteristics of patients with primary adrenal lymphoma. Patients We retrospectively reviewed the cases of five patients with primary adrenal lymphoma who were treated in our hospital between April 2004 and March 2015. We investigated the characteristics of the clinical and pathological findings, treatment, prognosis and complications of adrenal insufficiency. Results Adrenal insufficiency, which was confirmed by the laboratory data at the initial presentation, was observed in two cases. One case was complicated by relative adrenal insufficiency during a course of chemotherapy. The plasma adrenaline and urinary adrenaline levels were decreased in four cases and three cases, respectively. Diffusion MRI was radiologically diagnostic. In all of the cases, the patients were pathologically diagnosed with diffuse large-B cell lymphoma and were treated with rituximab and CHOP (cyclophosphamide, doxorubicin, vincristine and prednisone)-like chemotherapy. Two patients received central nervous system prophylaxis with high-dose methotrexate. Four of the patients survived and one patient died during the follow-up period. Conclusion The early detection of adrenal insufficiency and the administration of an appropriate dose of hydrocortisone are necessary during the course of chemotherapy as well as at the initial manifestation. The exclusion of adrenal dysfunction prior to invasive diagnostic procedures, such as CT-guided needle biopsy, is also critical.

Paroxysmal Hypertension Induced by an Insulinoma

Insulinoma is a rare, usually benign, pancreatic neuroendocrine tumor. The clinical features of an insulinoma are fasting hypoglycemia with neuroglycopenic symptoms including confusion and unusual behavior, while hypertension is usually not associated with the disease. We herein report a patient with insulinoma who manifested paroxysmal hypertension and neuroglycopenic symptoms. The possible etiology of hypertension induced by an insulinoma is catecholamine release in response to hypoglycemia, which may cause acute hypertension through activation of the sympatho-adrenal system. This case implies that sustained hyperinsulinemia due to insulinoma can be functionally linked to the induction of paroxysmal hypertension.

Diffuse large B-cell lymphoma involving the skeletal muscle

A 65-year-old man was referred to our hospital with extensive swelling of the left leg (Picture A). His serum levels of lactate dehydrogenase and soluble interleukin-2 receptor were elevated to 1,000 IU/L (reference range, 120-240 IU/L) and 3,922 U/mL (reference range, 122-496 U/mL), respectively. Magnetic resonance imaging of the leg showed an enlarged left thigh composed of the quadriceps femoris muscles with a high-intensity area (Picture B, arrows) and the femoral bone, with heterogenous spots on T2 images (Picture C, arrows). Positron emission tomography-computed tomography identified high uptake of F-FDG (SUVmax, 13.25), predominantly in the muscles of his left thigh and also in the supraclavicular, subclavicular, and abdominal lymph nodes (Picture D). Following a biopsy of the left su-