Intermittent chyluria in a young man

Chyluria is the passage of chylus into urine resulting in fistulization through the lymphatic system and the urinary system. This rare condition is usually caused by filaria infestation or malformations, neoplasia or trauma. We report a case of a 18-year-old man. The patient presented milky urine which had appeared after angiography following minor leg trauma. Physical examination revealed asymmetry of the face and cutaneous dyschromia. Blood tests revealed hypogammaglobulinemia and altered CD4/CD8 ratio (0.6). Urine tests showed proteinuria (30 mg/dl), lipiduria (triglycerides 750 mg/dl) and density of 1025. Renal function was normal. Abdomen computed tomography and urography were normal. Cystoscopy revealed the presence of milky urine in the bladder and selective catheterization revealed that the origin was the right ureter alone. Ascendent pyelography did not reveal any malformation of the urinary tract; but after this the chyluria spontaneously disappeared. The patient was rehospitalized 3 months later for recurrence. Lymphography was then performed and revealed a dilated lymphatic network with minute lacunar images projecting into the right kidney. Chyluria again disappeared spontaneously and recurred sporadically over the next two years in a patient who remained in good physical condition. The etiology of chyluria in a patient without filaria infestation is problematic, particularly when the most common causes (tuberculosis, neoplasia, trauma) are excluded as in our case. The asymmetry of the face, together with cutaneous dyschromia and the presence of a subarachnoidea cyst in the right temporal region suggested our patient had multiple congenital malformations.     

Florid cemento-osseous dysplasia in a young Chinese man. Case report

Florid cemento-osseous dysplasia refers to a group of fibro-osseous lesions which are exuberant, multiquadrant and arise from the tooth-bearing area of the jaws. It is classically described as a condition occurring almost exclusively in middle-aged black women. A case of florid cemento-osseous dysplasia occurring in a young Chinese male is reported which was rare in regard to race and sex. This 20 year old Chinese man presented with huge symmetrical bony lesions in all four quadrants of the jaws. Clinical presentation, radiological findings and histological features of the excised specimens are described. Treatment of the lesions was unusual. Curettage was first done with minimal benefit and it was followed by mandibular recontouring to improve facial appearance. The outcome of these procedures will be discussed.     

Cutaneous purulent aspergillosis in a young man with chronic granulomatous disease

We describe a case of cutaneous purulent aspergillosis in a 19-year-old man with chronic granulomatous disease (CGD) treated successfully with a 6-month regimen of itraconazole. The therapeutic effect of the drug was seen after 1 month of administration. Surgical treatment of the skin lesions, although first planned, was not necessary.     

Pneumomediastinum: an unusual complication of bronchial asthma in a young man

We report a patient with bronchial asthma who presented with pneumomediastinum, pneumopericardium, pneumoretroperitoneum, pneumorrhachis and extensive subcutaneous emphysema, after a period of coughing. Pathogenesis, diagnostic procedures and treatment of pneumomediastinum and its complications are discussed.     

Tuberculosis presenting with generalized lymphadenopathy, pulmonary infiltrates, and bone destruction in a young man

Although tuberculosis is still relatively common in Canada, particularly among recent immigrants, one tends not to consider it very strongly in the absence of definite pulmonary symptoms and in the face of negative smears of expectorated sputum and bronchial washings. We present the case of a young immigrant from Ethiopia who presented with mild constitutional symptoms, bony tenderness of the chest wall, generalized lymphadenopathy, and rib erosions--a set of features which, in the face of smear-negative sputum and bronchial washings, raised a possibility of lymphoma. Eventually, diagnosis of tuberculous lymphadenitis and osteomyelitis was established and the patient responded well to appropriate therapy.     

A 78-year-old man with young onset parkinsonism and sudden death

We report a right-handed 78-year-old man with early onset parkinsonism. The patient had an onset of micrographia at 23 years of the age in 1939. Seven years later he started to drag his right foot, and at 38 years of age, he walked with small steps with festination. Tremor was also present in his right hand. His daily life was independent as a otorhinolaryngologist. He visited our clinic on March 24, 1977 when he was mentally sound and showed mild parkinsonism consisting of masked face, stooped posture, small step gait, bradykinesia, and right side dominant rigidity and tremor. He showed good response to trihexyphenidyl and amantadine HCl. Two month later, he developed dyskinesia and some worsening of parkinsonism, and was admitted to our hospital for the first time. He was treated with 400 to 600 mg/day of levodopa/ carbidopa. He showed marked improvement, however, dyskinesia remained in his mouth. He was doing well until 77 years of age (June of 1993) when he developed hallucination and motor fluctuations. He was admitted again to our hospital on June 22, 1993. On admission, he was alert and appeared mentally sound. However, Hasegawa dementia scale was 18/30. Upward gaze was slightly restricted (3/5). Voice was somewhat small but no masking was noted. His posture was stooped and the gait was of small step. Dyskinesia was noted during walk. No rigidity nor tremor was noted. Deep tendon reflexes were lost but no sensory loss or motor weakness was noted. Routine laboratory studies were unremarkable. A cranial CT scan revealed only mild to moderate cortical atrophy. Motor and sensory conduction velocities were within normal limits, however, motor action potentials could not be obtained with stimulation to the right common peroneal nerve. He was treated with 600 mg/day of levodopa with carbidopa, 100 mg of amantadine HCl, 300 mg of Dops, and 25 mg of tiapride. He continued to show motor fluctuations, and was discharged on July 23, 1993. Since then his motor functions had become progressively worse with frequent falls, but he was still able to walk without support. On October 3 of 1994, he went to bed as usual. On the next morning, he was found dead in his bed at 9: 30. The patient was discussed in neurological CPC, and the chief discussant arrived at the conclusion that the patient had young-onset Parkinson's disease with Lewy bodies in the substantia nigra. Opinions were divided between Parkinson's disease and Lewy body negative young onset parkinsonism. Postmortem examination revealed obstruction of the trachea by aspirated foods, and the cause of death appeared to have been suffocation by the foods. Macroscopically, the external appearance of the brain was unremarkable except for slight frontal atrophy. The substantia nigra showed depigmentation in the lateral part, but the pigmentation of the medial part was well preserved. Upon histologic examination, the number of pigmented neurons in the dorsomedial part was well preserved. In the lateral part, pigmented neurons were well preserved in the dorsal area, however, in the ventral area, only non-pigmented neurons were seen; they appeared to be neurons in the pars reticulata. No gliosis was seen in any of the nigral areas. No Lewy bodies were seen in the remaining neurons. So-called immature neurons with rounded shape without neuromelanin could not be detected. The locus coeruleus neurons were well preserved. The putamen and the other basal ganglia structures were also intact. Slight myelin pallor was noted in the subcortical white matter, however, otherwise cerebral cortices were normal. The histology of this patient is unique in that only the ventrolateral part of the substantia nigra showed abnormal finding consisting of lack of pigmented neurons without gliosis. It is not clear whether the nigral change represents degeneration or a congenital "hypoplasia'. To our knowledge, such a unique pathology of the substantia nigra has not been reported in the literature. Our patient ma     

A surgical case of liposarcoma in the chest wall within the thoracic cavity of a young patient

A 21-year-old man presented to our hospital on June 7, 1990, asymptomatic but with an abnormal shadow in his chest roentgenogram. A well-demarcated tumorous shadow was seen in the left hilum of the lung in the frontal view of the chest X-ray, and adjacent to the posterior chest wall in the lateral view. A percutaneous needle biopsy was performed, and since there were no malignant findings, the tumor was left untreated for future examinations. However, since the tumor was observed to be enlarging gradually in subsequent chest X-rays, it was resected surgically on June 18, 1991. The tumor was fist-sized and its surface smooth, and was protruding into the thoracic cavity. At surgery, the tumor was separated from the chest wall. The tumor, weighing 170 grams, was diagnosed pathologically as a liposarcoma (myxoid type). It was confirmed that there was no infiltration into the surrounding tissue and that total resection had been performed. There are few reports of liposarcomas within the thoracic cavity developing in the extra-mediastinal chest wall. As our case is the second in Japan occurring in a young patient, we present it here together with a review of the literature.