Megaesophagus and pneumonia associated with Mycobacterium chelonei. A case report and a literature review

The case of a 37-year-old woman who developed a subacute, bilateral, noncavitary pneumonia 5 years after a colon interposition esophagoplasty is presented. Mycobacterium chelonei, subspecies abscessus, was assigned a pathogenic role based on the findings of (1) a clinical and roentgenographic picture consistent with tuberculosis, (2) sputum smears showing acid-fast bacilli, (3) repeated sputum cultures yielding heavy growths of Mycobacterium chelonei, subspecies abscessus, and (4) a 12-mm by 12 mm-skin test response to homologous antigen (purified protein derivative-CL) with no response to an equivalent dose of purified protein derivative-S. The patient recovered fully without significant antituberculous chemotherapy. A survey of the literature revealed 11 similar case reports featuring a documented association between megaesophagus and pulmonary infection with rapidly growing mycobacteria.     

Wilms tumor associated with polycythemia: case report and review of the literature

A 13 year review of patients diagnosed to have Ludwig's angina admitted to the Christian Medical College and Hospital, Vellore, India, between March 1982 and April 1995 is presented. The patients were either admitted to the ENT or paediatric surgical units. There were 41 patients, 24% being children and 76% adults. The clinical profile and outcome of these two groups were compared. In the paediatric group, none had dental caries while in the adult group, 52% had associated dental caries. In children, 70% were controlled with conservative medical management unlike the adults of whom 81% needed incision and drainage. Tracheostomy was necessary in 10% of the children and in 52% of the adults. The mortality was 10% in both groups.     

Granulomatous orchitis. A case report and review of the literature

We developed a method for determination of motor conduction along the mandibular and sensory conduction along the lingual and inferior alveolar nerves in 10 controls and 6 patients with lingual neuropathy following lower wisdom tooth extraction. Patients with lingual neuropathy had reduced/absent or delayed compound sensory action potentials and normal conduction along the fibers of the inferior alveolar nerve and mandibular nerve. The method provides a useful electrophysiological means of evaluating lingual nerve lesions.     

Cirrhosis and pregnancy. A case report and review of the literature

A clinical case of a pregnant suffering from hepatic cirrhosis with ascites, splenomegaly and portal hypertension is described. The pregnancy carried on till the 31st week, even though with the repeated use of tocholytic agents. Cesarean section was performed because of the onset of serious jaundice and the decline of general maternal conditions. The infant, who had an Apgar score of 8 at the 1st and 5th minute, died on the 10th day because of accuse haemorrhagic interstitial pneumonitis in premature lungs and hepatopathy associated with widespread jaundice. The mother was discharged on the 25th day of the postpartum period, in light of the net improvement of her general metabolic condition, the sudden regression of the jaundice and the decrease of the cholestasis indices. A review of the literature discussing maternal complications fetal risks, management of pregnancy and delivery and outcome of the newborn are presented.     

A rare case of ovarian leiomyoma. A case report with review of literature]

We report an exceptional case of ovarian leiomyoma in 73 years old patient. Leiomyoma of the ovary is a very rare tumor. Most cases are asymptomatic, this benign neoplasm is usually found incidentally on routine pelvic examination, at surgery or at autopsy. The available literature is reviewed. The possible histogenesis of ovarian leiomyoma was discussed.     

Intrapericardial teratoma. A case report and a literature review

A case of newborn intrapericardial teratoma is reported. The clinical, echocardiographic, tomographic and histologic features are described, and also, the therapeutic options. The newborn was submitted to surgical excision of the intrapericardial tumor and has a clinical follow-up greater than four years.     

Extraskeletal retroperitoneal osteosarcoma. A case report and review of the literature

Extraskeletal osteosarcomas, localized in the retroperitoneal space, are very rare neoplasms (only 200 cases have been described); the case indicated is the second one observed in Literature. The extraskeletal osteosarcomas are very malignant mesenchymal neoplasms reproducing osseous and cartilaginous tissue disconnected from the skeletal system. From a histological point of view, the extraskeletal osteosarcomas are constituted of osseous and cartilaginous tissue with osteo-fibro-chondroblastic cells: the different representation of three types of cells define the variety of extraskeletal osteosarcomas (osteoblastic, chondroblastic, fibroblastic). Histological and ultrastructural study failed to find differences between skeletal and extraskeletal osteosarcomas. In the clinical history of some patients traumas or irradiation has been referred; although extraskeletal osteosarcomas etiology is frequently unknown. Diagnosis is generally delayed because symptoms are often absent of uncertain. Prognosis of these neoplasms is always very poor, and specially in relation to the dimension and to the possibility of its radical removal. All patients died until 2-3 years after surgery.     

MALT lymphoma of the parotid gland associated with Sj?gren syndrome. A case report and review of literature

Sj?gren syndrome (SS) is a chronic inflammatory process involving the exocrine glands. Its association with the development of lymphomas is well documented, and this risk has estimated to be 44 times that of the general population. A MALT lymphoma of the parotid gland in a patient with Sj?gren syndrome is reported. We review the literature and comment the clinical and histopathological features of these tumors.     

Septicemia due to Shigella. A case report and review of the literature

To develop a proper protocol for biological exposure monitoring of acetone, we evaluated whether exposure to acetone on the previous day affects the biological monitoring value at the end of a work day. One hundred and ten male workers exposed to acetone in three acetate fiber manufacturing plants were monitored using a liquid passive sampler on two consecutive working days after 2 days without exposure. Urine samples were collected at the start of the workshift and the end of the shift on both days for each subject. For ten exposed workers urine samples were collected approximately every 2 h during and after the first working day until the following morning. Acetone concentrations in urine (Cu) at the start of the first working day were 1.3 +/- 2.4 (range: ND-14.1) mg/l in nonexposed workers and 2.4 +/- 5.6 (range: ND-40.3) mg/l in exposed workers. The urinary acetone concentration at the beginning of the second working day indicated that urinary levels of acetone do not decline to background level by the following morning when exposure concentration exceeds 300 ppm. However, linear regression analysis demonstrated that the relationship between environmental exposure level and urine level was similar on the 1st day and the 2nd day. Thus, although urine acetone levels did not return completely to baseline after high exposures, under the present exposure levels the exposure on the previous day did not significantly affect urinary acetone at the end of the workshift of the next day.     

Primary tracheal non-Hodgkin's lymphoma. A case report and review of the literature

BACKGROUND: Primary tracheobronchial non-Hodgkin's lymphoma (NHL) is an uncommon occurrence. The authors report a patient who presented with primary tracheal NHL, the sixth such patient described in the literature. METHODS: Using a MEDLINE search, 41 additional patients presenting with symptomatic primary or secondary tracheobronchial NHL were identified. The characteristics, management, and outcome of these patients are described. RESULTS: Patients with NHL of the upper respiratory tract present with dyspnea, wheezing, and cough, and frequently are misdiagnosed as having asthma. The majority of patients have additional sites of intrathoracic disease with tracheobronchial involvement occurring in the setting of advanced or relapsed NHL. Low grade histology is seen most commonly in patients with primary tracheal NHL. Several patients demonstrate the typical histologic features of mucosa-associated lymphoid tissue. Surgery, chemotherapy, and radiation therapy have been used alone or in combination for treatment. The outcome of these patients does not appear different from that observed in patients with lymphomas of similar histology and stage that do not involve the tracheobronchial tree. CONCLUSIONS: Thoracic surgeons, pulmonologists, and oncologists should recognize that NHL can rarely be confined to the trachea or bronchi. NHL should be considered in the differential diagnosis of airway obstruction, because it represents a highly treatable malignancy.     

Congenital pseudoarthrosis of the radius. A case report and review of the literature

Congenital pseudoarthrosis of the radius (CPR) is rare. Only ten cases seem to have been found in the literature. Congenital pseudoarthrosis of the radius usually is associated with neurofibromatosis or fibrous dysplasia. This is a report of a congenital pseudoarthrosis of the left radius associated with congenital dislocation of the left hip in a five-month-old girl. Apart from the pseudoarthrosis, there were no visible abnormalities. This patient was treated successfully with excision of the pseudoarthrosis, shortening osteotomy of the ulna, grafting, and intramedullary fixation of both the radius and the ulna. Review of the literature disclosed the extreme difficulties in treating this condition. It was also found that vascularized fibular graft has been used successfully.     

Unusual metastasis of renal cell carcinoma. A case report with review of the literature

BACKGROUND: The occurrence of metastasis of renal cell carcinomas in the head and neck region is extremely rare. Metastasis in the larynx, hypopharynx, and the nasal sinuses has been reported. We report here about a 55-year-old female with metastasis in the soft palate and tonsil, which occurred 10 years following tumor nephrectomy. RESULTS AND CONCLUSIONS: The incidence of metastasis in renal cell carcinomas can be observed even many years following initial curative treatment of the primary tumor. Although rare in the region of the head and neck, they can often be mistaken for benign tumors such as hemangiomas or inflammatory tissue. The treatment of choice is radical surgical resection.     

Veno-occlusive disease of the liver associated with oral contraceptives: case report and review of literature

This report concerns a young woman who, after taking a contraceptive preparation orally for three years, developed the Budd-Chiari syndrome as the result of a widespread chronic obliterative process involving the intrahepatic efferent venous system. Her prolonged course, which failed to respond to an end-to-side portacaval shunt procedure, mimicked chronic hepatitis evolving to cirrhosis. Additional noteworthy features were the presence of two small benign hepatic adenomas, observed both at biopsy and at necropsy, a lesion recently recognized as a complication of anovulatory drugs, and widespread hepatic calcifications found at autopsy.     

Brown tumor in association with secondary hyperparathyroidism. A case report and review of the literature

The anti-inflammatory agent D-myo-inositol-1,2,6-trisphosphate (1,2,6-IP3) has shown beneficial effects in experimental burns following systemic administration. The purpose of this study was to investigate the effect of topical 1,2,6-IP3 cream on a standardised full-thickness 1 cm2 burn injury in rats. The experimental cream contained a transcutaneous absorption enhancer, hexylbetaine. Five different treatment groups were used. Two experimental groups of burned rats received either 1,2,6-IP3 cream with hexylbetaine (n = 10) or without hexylbetaine (n = 10). Two burned control groups were treated either with hexylbetaine cream (n = 10) or placebo cream (n = 10), while a third control group was untreated (n = 14). The various creams (0.5 g) were administered to the experimental burn area and allowed to remain for 3 h covered with an occlusive dressing. Spectrophotometrical quantification of Evans blue albumin extravasation was used to evaluate the effect of the experimental creams on vascular permeability following the burn trauma. Results showed a significant reduction of albumin extravasation both by 1,2,6-IP3 (p<0.05) and by hexylbetaine alone (p<0.01), as compared to placebo cream-treated animals. The transcutaneous absorption enhancer hexylbetaine did not further improve the effect of 1,2,6-IP3 on burn oedema. In conclusion, both topical 1,2,6-IP3 and hexylbetaine induced a significant reduction of albumin extravasation in burned skin. The effect of 1,2,6-IP3 could be related to previously shown anti-inflammatory actions of the agent, while the mechanisms of actions of hexylbetaine remain to be investigated.     

Acquired Glanzmann's thrombasthenia associated with Hodgkin's lymphoma: a case report and review of the literature

BACKGROUND: Acquired Glanzmann's thrombasthenia is a rare hemorrhagic diathesis resulting from impaired adhesive function of the platelet receptor GPIIb/IIIa (alpha(IIb)beta3). Typically, this disorder develops during adulthood, with patients manifesting fluctuating clinical and laboratory findings. To date, the underlying defect of most if not all cases of acquired Glanzmann's thrombasthenia results from an autoantibody or plasma protein inhibitor directed toward a demonstrably normal GPIIb/IIIa glycoprotein. METHODS: In this report, a patient with a history of treated Hodgkin's lymphoma presented with a severe hemorrhagic diathesis characterized by mild thrombocytopenia, a prolonged bleeding time, and defective platelet aggregation. RESULTS: Examination of the patient's platelet GPIIb/IIIa by Western blot analysis revealed no abnormality. Mixing studies demonstrated a non-immunoglobulin G plasma inhibitory factor, whereas flow cytometry analysis revealed elevated platelet-associated immunoglobulin (Ig) M. After an emergency colectomy for severe hemorrhage, the patient's qualitative and quantitative platelet parameters significantly improved. Pathology of the resected colonic segment demonstrated atypical lymphoid hyperplastic lesions. CONCLUSIONS: To the authors' knowledge, this is the first reported case of acquired Glanzmann's thrombasthenia associated with a putative IgM autoantibody. Furthermore, this report verifies the association of acquired thrombasthenia with lymphoproliferative disease. Although rare, awareness of this hemorrhagic diathesis as a possible sequelae of active or treated lymphoid disorders should encourage clinical vigilance of these patients.     

Sonographically detected fetal and placental abnormalities associated with trisomy 16 confined to the placenta. A case report and review of the literature

Previous studies have demonstrated that cortical spreading depression (CSD) induces neuronal tolerance to a subsequent episode of ischemia. The objective of the present investigation was to determine whether CSD alters levels of mRNA coding for putative neuroprotective proteins. Unilateral CSD was evoked in male Wistar rats by applying 2 mol/L KCl over the frontal cortex for 2 hours. After recovery for 0, 2, or 24 hours, levels of several mRNA coding for neuroprotective proteins were measured bilaterally in parietal cortex using Northern blot analysis. Levels of c-fos mRNA and brain-derived neurotrophic factor (BDNF) mRNA were markedly elevated at 0 and 2 hours, but not 24 hours after CSD. Tissue plasminogen activator (tPA) mRNA levels were also significantly increased at 0 and 2 hours, but not 24 hours after CSD. Levels of the 72-kDa heat-shock protein (hsp72) mRNA were not significantly increased by CSD, except for a small elevation (20%) at 2 hours recovery. Levels of the 73-kDa heat-shock cognate (hsc73) mRNA were slightly, but significantly, increased at 2 and 24 hours of recovery. Finally, levels of mRNA for protease nexin-1 and glutamine synthetase were not significantly altered by CSD at any time studied. The current results support the hypothesis that neuronal tolerance to ischemia after CSD may be mediated by increased expression of FOS, BDNF, or tPA, but not by increased expression of hsp72, hsc73, nexin-1, or glutamine synthetase.     

Surgical management of osteochondritis dissecans of the capitellum

Ten cases of osteochondritis dissecans of the humeral capitellum which were treated surgically are reviewed. All 10 cases were males and involved the dominant side. The ages at surgery ranged from 13 to 17 years. Follow-up ranged from 1 to 7 years. All of the youths had competed in organized athletics, either baseball or football. By position there were three pitchers, two catchers, two infielders, and one outfielder; in addition there were one quarterback and one linebacker. Only one patient presented with locking of the elbow, whereas the others presented with pain and limitation of extension. The locked elbow was explored immediately and the others were explored after immobilization failed to relieve their symptoms. In seven of the joints a loose fragment of the capitellum was found lying either in the joint or in a defect in the capitellum. The fragment had multiple small holes. In three cases there was no loose fragment. In this situation a corticol window was cut above the capitellum. The capitellum was then drilled and bone was grafted from above. Over all, there were one excellent, six good, one fair, and two poor results. There seemed to be little difference between curretting alone or curetting and drilling. The cases with the cartilage intact and bone grafted from above did worse, with one fair and one poor result of three cases. The two poor results required further surgery, which consisted of partial excision of the capitellum. All cases lacked elbow extension before and after surgery, but nine of 10 gained some motion after surgery. Pre- and postoperative x-rays are shown in this report and a brief review of the literature concerning osteochondritis dissecans is presented.