A case of eosinophilic cellulitis associated with angioedema and eosinophilia / 천식및알레르기

Eosinophilic cellulitis was first described by Wells, which is characterized by cellulitis like skin lesion, eosinophilic infiltration of dermis and subcutaneous fat with characteristic flame figure appearance of chollagen bundles. Peripheral eosinophilia is not necessary for the diagnosis of eosinophilic cellulitis, but association with hypereosinophilic syndrome was reported. Episodic angioedema associated with eosinophilia was first described by Gleich et al. as recurrent attack of angioedema; peripheral eosinophilia; and body weight gain; they described this as separate disease entity rather than variant of the hypereosinophilic syndrome. Both angioedema associated with eosinophilia and eosinophilic cellulitis were reportedly associated with hypereosinophilic syndrome, and we believe both diseases are variants of idiopathic hypereosinophilic syndrome. We experienced a patient with eosinophilic cellulitis who has expressed skin lesion resembling angioedema associated with eosinophilia, and who also has involvement of pleura and gastrointestinal tract. We speculate that this patient represents an overlap syndrome of idiopathic hypereosinophilic syndrome, eosinophilic cellulitis and angioedema associated with eosinophilia.

Correlation between serum prolactin levels and immunocytochemical findings of pituitary adenomas in patients with acromegaly / 대한내과학회지

BACKGROUND: Acromegaly occurs by excessive secretion of growth hormone and more than 99% of cases are caused by a growth hormone-secreting pituitary adenoma. Pituitary adenomas expressing multiple immunoreactivities are common. We assumed that the pituitary adenomas which is immunochemically detected growth hormone and prolactin are responsible for it and reviewed 28 patients with acromagaly to determine the correlation between serum hormonal level and immunocytochemical finding. METHODS: Twenty-eight patients with acromegaly who underwent surgery of pituitary adenoma in Samsung Medical Center from October 1998 to may 2001 were included. Baseline hormonal evaluations and several endocine tests were performed. Immunocytochemical stain was done. RESULTS: According to the extent of hormonal stain, the adenoma was divided into two groups. The adenoma showing immunoreactivity over 50% to growth hormone was 100%, to prolactin was 71.4% and to FSH was 25.0%. The extent of other hormonal stain was less than 20%. There were no significant differences in age, sex, the ratio of macroadenoma and microadenoma, the basal serum GH level, serum IGF-1 level, and the response to TRH, somatostatin and bromocriptine suppression test between the two groups divided by the the extent of prolactin stain. But the serum prolactin level was 55.0+/-63.4 ng/mL, and 19.9+/-12.2 ng/mL each in two groups which was siginificantly increased in the adenoma showing immunoreactivity over 50% to prolactin. CONCLUSION: Acromegaly patients with higher expression of prolactin on immunocytochemical studies showed higher serum prolactin levels and patients with hyperprolactinemia showed higher serum IGF-1.

Occupational asthma induced by cephalosporin precursor / 천식및알레르기

A 41-year-old male pharmaceutical worker developed dyspnea and wheezing 10 years after beginning the work. He had handled powder of cephalosporin precursor, 7-aminocephalosporanic acid (7-ACA). He had suffered from dyspnea, chest tightness and wheezing in the workplace for the last three years. Recently, these symptoms were aggravated. His pulmonary function test showed severe obstructive pattern and PC(20) was 0.06 mg/ml on MBPT(methacholine bronchial provocation test). Skin prick test and intradermal test showed positive reactions to ceftriaxone, cefazolin, etc. During the skin test, he suffered from an asthmatic attack and anaphylactic shock. Bronchial provocation test with cefotiam elicited a dual asthmatic reaction. We presumed that IgE-mediated mechanism may be mainly involved in the pathogenesis of cephalosporin induced occupational asthma, in addition to other immunologic or non-immunologic mechanisms. Further studies will be necessary to investigate the pathogenesis of this disease.

Diffuse nesidioblastosis underwent reoperation after streptozotocin treatment / 대한내분비학회지

Nesidioblatosis is a term that describes small clusters of pancreatic islet cells budding off exocrine ducts, and is commonly reported in infants with intractable idiopathic hypoglucemia. The onset of nesidioblastosis in adults is an extremely rare entity associated with hypersecretion of insulin and the treatment of choice is pancreatic resection. Medical treatment, including somatostatin, propranolol, diazoxide, hydrochlorthiazide and streptozotocin have achieved limited success. We experienced a case of adult nesidioblastosis that underwent reoperation after the failure of medical treatment following an inappropriate first operation. A 54-year old man was admitted due to intermittent hypoglycemic symptoms, which had been relieved by carbohydrate ingestion. Hyperinsulinemic hypoglycemia was documented during a prolonged fast. Image studies found no localized lesion, so a distal pancreatectomy was performed. The pathological examination of the resected pancreas revealed irregularly sized islets and a scattering of small endocrine cell clusters throughout the acinar tissue and ductuloinsular complex. After a partial pancreatectomy the hypoglycemia had not disappeared. The patient did not want to undergo a reoperaton due to the post operative wound infection that occurred after the distal pancreatectomy. Therefore, diazoxide, somatostatin, propranolol, and streptozotocin was used as the alternative to an operation. However, the hypoglycemia persisted during and after the medical treatment. Finally, he underwent a near total pancreatectomy (85%), and the hypoglycemia disappeared. The extent of pancreatectomy is important in clinical outcome of patients with nesidioblastosis

Successful treatment of refractory hemolytic uremic syndrome with intravenous gamma-immunoglobulin therapy / 대한내과학회지

The hemolytic uremic syndrome (HUS) is a clinical syndrome defined by the presence of thrombocytopenia, microangiopathic hemolytic anemia and acute renal failure with or without a clinically apparent etiology. The conventional treatment of choice is plasmapheresis as a first-line therapy. Most patients respond to plasmapheresis whereas some patients are refractory to the therapy. The second-line therapy is not well established although various therapies such as steroid, vincristine, intravenous immunoglobulin have been suggested. The intravenous immunoglobulin therapy in refractory hemolytic-uremic syndrome have rarely been successful in complete remission. We report a case of refractory HUS in a 48 year-old man who developed hemolytic anemia, thrombocytopenia, acute renal failure and ischemic retinopathy. The patient was refractory to plasmapheresis as a first-line therapy. The patient received intravenous immunoglobulin therapy as a second-line therapy after 8 days of plasmapheresis, which subsequently resulted in a complete remission of refractory HUS. The complete remission using immunoglobulin in HUS has not been previously reported in Korea.