Granulomatous(lobular) mastitis is a distinct disease entity of unknown etiology which is characterized by noncaseating granulomatous lobulocentric inflammation. We describe a rare case of granulomatous(lobular) mastitis of a 36 year-old pregnant woman a review of the literature. The mass which was discovered in the third month of her pregnancy, began as a localized, nontender mass on the left breast and persisted during her entire pregnancy. It decreased slightly in size when she began taking post-partum bromocriptine. Clinically and mammographically, the mass was highly suspected as a carcinoma with axillary lymph node metastasis. Fine needle aspiration smears revealed numerous aggregates of granulomas composed of epithelioid histiocytes admixed with multinucleated giant cells of Langhans' and foreign body type, and collections of polymorphonuclear leukocytes. Ziehl-Neelsen, silver methenamine and PAS stain were negative for acid-fast bacilli, fungus, and bacilli on the smear respectively. Histologically, granulomatous inflammation was centered on the breast lobules. Caseation necrosis was absent, instead, numerous microabscesses were formed in the center of the granulomas. Cultures of the fresh tissue for the AFB, aerobic and anaerobic bacteria, and fungus were all negative. Excision of the mass was performed without further treatment and there was no recurrence of the mass 6 months postoperatively. An autoimmune mechanism, infection, and some association with oral contraceptives have been suggested as etiologic factors in the literature.
Pregnancy ; Female ; Humans

We report a case of secretory carcinoma with axillary lymph node metastasis in a 21-year old woman. She was aware of a mass in her breast for 10 years and noticed a rapid growth of the preexisting mass during the last years. Histologically, the tumor was composed of micropapillary and microcystic or cribriform glandular structures which contained eosinophilic, mucinous, intraluminal secretions. The center had a dense hyalinized strama with a solid infiltrative growth of tumor cells with intracytoplasmic secretory vacuoles at the periphery. In addition, marked intraductal papillary epithelial proliferations were present at the superficial portions of the tumor near the nipple. Prognostic factors and their relationship to juvenile papillomatosis are discussed with a review of the literature.
Female ; Humans ; Neoplasm Metastasis ; Breast Neoplasms

The congenital cystic adenomatoid malformation (CCMA) of the lung is a rare variant of congenital cystic lung disease consisted of one or usually multiple interconnecting cysts in the pulmonary parenchyma lined by cuboidal and columnar epithelium. This diease present clinically in three ways: 1) stillborn or perinatal death, 2)progress respiratory distress in the newborn, and 3)acute and chronic pulmonary infections in the older infant and child. The onset of symptoms, which are cyanosis, tachypnea, and other forms of respiratory distress, usually occurs at or shortly after birth, This manifestations are related to compression of the remained normal ung by expansion of the cysts. We have experienced a case of congenital cystic adenomatoid malformation of the lung in a 1-day-old male infant who had tachypnea. A right upper lobectomy was done with satisfactory postoperative courses clinically and radiologically at 8 months of age. A brief review of the related literature is presented.
Child ; Cyanosis ; Cystic Adenomatoid Malformation of Lung, Congenital* ; Epithelium ; Humans ; Infant ; Infant, Newborn ; Lung Diseases ; Lung* ; Male ; Parturition ; Tachypnea

Acute suppurative thyroiditis is a rare because of high resistance to bacterial infection of thyroid gland, which is rich vascularity and lymphatic drainage, a high iodine content and complete encapsulation. The common clinical manifestations are fever, neck pain and localized mass of thyroid area. This thyroiditis is more common in left thyroid lobe. The most important causal microorganism are staphylococci and streptococci, with frequent isolation of mixed flora and anaerobes. Diagnosis was easily made by typical clinical manifestation, ultrasonography, thyroid imaging and fine needle aspiration. Treatment usually consist of appropriate antibiotic therapy and surgical drainage when abscess formation develops. We report a typical case of acute suppurative thyroiditis in 1 year old female infant with brief review of literatures.
Abscess ; Bacterial Infections ; Biopsy, Fine-Needle ; Diagnosis ; Drainage ; Female ; Fever ; Humans ; Infant ; Iodine ; Neck Pain ; Thyroid Gland ; Thyroiditis ; Thyroiditis, Suppurative* ; Ultrasonography

PURPOSE: To describe the radiological differences between tuberculous(TBB) and non-tuberculous bronchiectasis(NTBB). MATERIALS AND METHODS: Chest radiographs(n=62), bronchograms(n=18), and CT scans(n=52) of 37 patients with TBB and 25 patients with NTBB were reviewed retrospectively. Diagnostic basis for TBB were positive sputum AFB with or without history of anti-tuberculous chemotherapy(n=35), and radiological findings of pulmonary tuberculosis (n=2). Four of NTBB had a history of severe respiratory tract infection in childhood. RESULTS: Air-fluid levels on chest radiographs were seen in 2% of TBB, and 20% of NTBB. On bronchograms, all patients with TBB had combined focal bronchostenosis, whereas patients with NTBB had tubular(50%), cystic(17%), or mixed(33%) pattern of dilatation without stenosis. On CT scans, focal emphysema was seen in 86% of the patients with TBB, and 38% of the patients with NTBB. Peribronchiolar infiltration were seen in 78% and 44% of patients with TBB and NTBB, retrospectively. CONCLUSION: Basic radiological difference between TBB and NTBB was that the former had coexistent sten.
Bronchiectasis* ; Constriction, Pathologic ; Dilatation ; Humans ; Pulmonary Emphysema ; Radiography, Thoracic ; Respiratory Tract Infections ; Retrospective Studies ; Sputum ; Thorax ; Tomography, X-Ray Computed ; Tuberculosis, Pulmonary

46,XX male is a rare abnormality of sex determination with an incidence of 1 in 20,000 male neonates. The clinical manifestations of 46,XX males are usually hypogonadism, gynecomastia, azoospermia, and hyalinations of seminiferous tubules, with altered hormonal levels at puberty. Less frequently, some sexual ambiguities are found, always with sterility owing to reduced testicular development. The origin of male phenotype in 46,XX male could be the results of at least three different mechanisms:translocations of Y sequence, including the SRY gene, to an X chromosome or to an autosome(about 90% of cases); a mutation in a yet unknown X-linked or autosomal gene in the testis-determinating pathway, and cryptic Y chromosome mosacism. We experienced a case of SRY-positive 46,XX male in a 21-year-old man with small testes. (J Korean Soc Pediatr Endocrinol 2003;8:184-188)
Adolescent ; Azoospermia ; Genes, sry ; Gynecomastia ; Humans ; Hyalin ; Hypogonadism ; Incidence ; Infant, Newborn ; Infertility ; Male* ; Phenotype ; Puberty ; Seminiferous Tubules ; Testis ; X Chromosome ; Y Chromosome ; Young Adult

46,XX male is a rare abnormality of sex determination with an incidence of 1 in 20,000 male neonates. The clinical manifestations of 46,XX males are usually hypogonadism, gynecomastia, azoospermia, and hyalinations of seminiferous tubules, with altered hormonal levels at puberty. Less frequently, some sexual ambiguities are found, always with sterility owing to reduced testicular development. The origin of male phenotype in 46,XX male could be the results of at least three different mechanisms:translocations of Y sequence, including the SRY gene, to an X chromosome or to an autosome(about 90% of cases); a mutation in a yet unknown X-linked or autosomal gene in the testis-determinating pathway, and cryptic Y chromosome mosacism. We experienced a case of SRY-positive 46,XX male in a 21-year-old man with small testes. (J Korean Soc Pediatr Endocrinol 2003;8:184-188)
Adolescent ; Azoospermia ; Genes, sry ; Gynecomastia ; Humans ; Hyalin ; Hypogonadism ; Incidence ; Infant, Newborn ; Infertility ; Male* ; Phenotype ; Puberty ; Seminiferous Tubules ; Testis ; X Chromosome ; Y Chromosome ; Young Adult

BACKGROUND: The toxicity of paraquat has been known to be caused by oxygen free radicals which leads to the lipid peroxidation and multiple organ failure. Although vitamin C has been known to be a potent antioxidant, recently there are numerous data which have shown that a low dose of vitamin C may act as a prooxidant due to the stimulation of the Fenton reaction with metal ions, which produces hydroxyl radicals. It has been reported that a deferoxamine in paraquat intoxication could reduce the production of the hydroxyl radicals by the inhibition of the Fenton reaction through the reduction of iron ion in tissue. The aim of this study was to evaluate the effect of the high and low dose of vitamin C and deferoxamine on lipid peroxidation and plasma TNF-alpha in paraquat intoxication. METHODS: Female Sprague-Dawley rats were divided into seven groups: control group which was not given paraquat(20mg/kg), P group which was given paraquat only, PVH group given paraquat and high dose of vitamin C(100mg/kg), PVL group given paraquat and low dose of vitamin C(10mg/kg), PVHD given paraquat, high dose of vitamine C and deferoxamine(100mg/kg), PVLD given paraquat, low dose of vitamin C and deferoxamine, and PD given paraquat and deferoxamine. Animals were killed at 6 and 24 hours after treatment. Malondialdehyde(MDA), superoxide dismutase(SOD) and glutathione(GSH) contents, catalase activity, plasma TNF-alpha, and histologic changes in the lung and liver tissue were measured. RESULTS: The lung histology in the PVH and PD or PVHD groups showed the significant decreases in the alveolar edema and interstitial thickness compared to the P group. The liver histology in the PVH and PVHD groups demonstrated marked differences in the central venous and sinusoidal dilatation compared to that of the P group. While the MDA levels of the lung and liver in the PVH and PD groups showed the significant reduction compared to that of the P group at 6 hours after treatment, all groups showed the significant changes compared to the P group at 24 hours. There was no significant change of the SOD levels of the lung and liver at 6 hours among all groups. At 24 hours, the SOD levels of the lung in PVH, PVL, and PVHD groups showed the significant increases compared to the P group. The increase of the SOD level in groups combined with deforoxamine, however, revealed a little reduction. The SOD level of the liver in PVH group only significantly increased compared to the P group at 24 hours. There was no significant change of the GSH level of the lung and liver among all groups at 6 hours. At 24 hours, the GSH level of the lung and liver were significantly increased in both PVH and PD group and PVH group, respectively, compared to the P group. Although the catalase activity of the lung was not significantly increased, that of liver was significantly increased in both PVHD and PD groups compared to the P group at 6 hours. The catalase activities of the lung and liver were significantly increased in PVH, PD, and PVHD at 24 hours. The concentrations of the Plasma TNF-alpha were slightly decreased at 6 hours and slightly increased at 24 hours compared to that of the P group, but they were not significant. CONCLUSION: This study showed that although the low dose of vitamin C had no effect, the high dose of vitamin C revealed a decrease of the MDA level and an increase of SOD, GSH, and catalase activity in the lung and lung and liver tissues, and the effect of the high dose of vitamin C increased with time. The administration of the deferoxamine with or without high dose of vitamin C, however, significantly showed the inhibition of the lipid peroxidation and antioxidant effect and low dose vitamin C decreased the effect of deferoxamine. The effects of the vitamin C and deferoxamine on plasma TNF-alpha were not clearly shown.
Animals ; Antioxidants* ; Ascorbic Acid* ; Catalase ; Deferoxamine* ; Dilatation ; Edema ; Female ; Free Radicals ; Humans ; Ions ; Iron ; Lipid Peroxidation* ; Liver ; Lung ; Multiple Organ Failure ; Oxygen ; Paraquat* ; Plasma ; Rats* ; Rats, Sprague-Dawley ; Superoxides ; Tumor Necrosis Factor-alpha ; Vitamins*

There have been no case reports of cranial nerve paralysis from total spinal anesthesia. This case report is to explain the possibility of cranial nerve involvement from high spinal anesthesia. A 17-year-old girl with a huge abdominal mass underwent exploratory laparotomy under spinal anesthesia. A lumbar tapping was performed in a sitting position at a level between L4-L5 using a 22 gauze pencil point needle, then 4 cc of 0.4% tetracaine in 6% D/W was injected into the subarachnoid space, followed by the patient lying down in a slight Trendelenburgs position. Shortly after the spinal injection of pontocaine, all signs of a high spinal block could be observed clearly, accompanided by the following increasing signs of intracranial nerve paralysis: phrenic nerve, vagus nerve, glossopharyngeal nerve, trigeminal nerve, trochreal nerve and finally oculomotor nerve in that order. She was then intubated and her respiration was controlled without delay. The patient recovered gradually in the reverse order one hour from the time after the spinal injection of pontocaine, without any permanent damage noted. Differential diagnosis between the shock and pure total spinal anesthesia are described here in.
Adolescent ; Anesthesia, Spinal* ; Cranial Nerves* ; Deception ; Diagnosis, Differential ; Female ; Glossopharyngeal Nerve ; Humans ; Injections, Spinal ; Laparotomy ; Needles ; Oculomotor Nerve ; Paralysis* ; Phrenic Nerve ; Respiration ; Shock ; Subarachnoid Space ; Tetracaine ; Trigeminal Nerve ; Vagus Nerve

The development of endometrial stromal sarcomas (ESSs) in foci of endometriosis is extremely rare, and few cases have been reported in the literature to date, particularly with regard to multiple extrauterine ESS. Here we report a case of endometrial stromal sarcoma with multiple metastasis that arose from an ovarian endometriotic lesion. The literature is also briefly reviewed.
Endometriosis ; Female ; Neoplasm Metastasis ; Ovary ; Sarcoma, Endometrial Stromal