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Four patients with granulomatous disease of the anterior visual pathway presented with optic neuropathy between July 1986 and February 1987, secondary to an orbital pseudotumor (n = 1) and sarcoidosis (n = 3). All but one became resistant to corticosteroid therapy, and all were subsequently treated for 1 to 2 years with low-dose cyclosporine alone or in combination with tapering doses of prednisone. Two patients experienced pronounced recovery of visual function, and visual function was stabilized in the other two; in one of these, magnetic resonance imaging showed substantial regression of intracranial lesions. Steroid-induced complications of uncontrolled hyperglycemia (n = 2) and obesity (n = 4) were controlled with cyclosporine. The authors conclude that low-dose cyclosporine is a safe and effective therapeutic alternative in ocular granulomatous diseases.
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PMID:Low-dose cyclosporine therapy of granulomatous optic neuropathy and orbitopathy. 180 Sep 35

Sarcoidosis can involve any tissue in the body and, consequently, the disease presents to clinicians of many different disciplines. We report two cases of sarcoidosis involvement of the nervous system: one male patient with right facial nerve palsy and a cutaneous involvement (supraorbital subcutaneous nodule) associated with bilateral hilar adenopathy; and a female patient presenting a hypothalamus involvement with diabetes insipidus, obesity, lethargy, sleep disturbances and amenorrhoea. In the later case an upper respiratory tract involvement was also present with nasal and tonsils sarcoidosis. A review of the clinical manifestations of neurosarcoidosis and the criteria for establishing the diagnosis is presented.
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PMID:[Neurosarcoidosis. Comments on 2 cases]. 210 Aug 66

In sarcoidosis and other granulomatous non-caseous diseases, the election treatment is immunosuppressive, mainly with cortisones that ensure more than 70% lasting remissions. Continuous use of cortisones for a long time (8-30 months) in high doses leads to serious side effects: gastric and intestinal ulcers, obesity, osteoporosis, suprarenal dysfunction, sensitivity to infections. Good results and elimination of the important side effects were obtained by treatment with Reprimum--a semisynthetic antibiotic with a wide spectrum and immunosuppressive properties--administered alone or with prednisone in small doses (15-20 mg once) in 6 weeks' series: 2 weeks--Reprimum 10/mg/kg daily +/- prednisone and for other 4 weeks--Reprimum 15 mg/kg twice a week +/- prednisone followed by two weeks' break. In 75 patients with histopathologically confirmed sarcoidosis (of whom 7-9.3% with outside-the-lung situs, too), the treatment with Reprimum gave: 94.7% lasting remission, only 5.3% failures, reduction of the treatment period to 6-12 months and the absence of any important side reaction. In other 37 sarcoidosis cases, failures of cortisone therapy (of which 11-30% relapses after 2-6 years), the treatment with Reprimum together with prednisone allowed recovery of 29 patients (78.4%). The same treatment with Reprimum, used in 22 patients with immunosuppressive treatment indication (dermatomyositis, Kaposi's syndrome, thrombocytopenias, nodose periarteritis, silicosis), of whom 18 (81.8%) were failures of the cortisone therapy, healed 20 of these cases (90.9%). Reprimum immunosuppressive property acts at the level of T4+ lymphocyte, involved in sarcoidosis pathogenesis. The functional blockage of T4+ lymphocyte can be also achieved by cyclosporine A.(ABSTRACT TRUNCATED AT 250 WORDS)
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PMID:[The advantages of Reprimum therapy in pulmonary sarcoidosis and other granulomatous diseases]. 255 63

Seven patients with advanced forms of mediastino-pulmonary sarcoidosis (involvement of the pulmonary parenchyma, and restrictive respiratory syndrome), confirmed by histopathologic investigation, have been treated with a new SV-rifamycin derivative, 1246-EH, or "Reprimum" without any other association. Reprimun was given in daily doses of 10 mg/kg of body weight for a period of two weeks, then, for another 4-5 weeks, the drugs was given intermittently in doses of 15 mg/kg of body weight twice weekly. Between two cycles of therapy a period of 14 days was free of drug administration. In all patients the treatment with the new derivative was well tolerated. The treatment was not followed by adrenal deficiencies, neither was noted obesity, bone decalcification, gastric ulcers, etc., which are commonly associated with prolonged corticoid therapy. The treatment with "Reprimun" efficient for all patients. A clear clinical and X-ray improvement was noted after the first three months of treatment with the new drug. Respiratory function, as well as clinical and radiological signs improved, the serum immunologic parameters, and the biochemical ones were also significantly improved (Ig, C3 total serum proteins and gammaglobulins). All parameters which were tested were finally improved and became normal. In only two of the seven patients the radiological aspects of the lung suggested the presence of sequellae at the end of the treatment. In these two patients there was a persistant reduction in the respiratory volume, as well as a discret hypoxemia. However, both these patients had a more advanced form of the disease, with extensive pulmonary fibrosis, and in these cases prolonged corticoid therapy which had previously been applied had failed. Reprimun had an immunological-modulatory effect in all patients, resulting in normal levels of IgG, IgA, and C3 in the first three months of therapy. There was a direct correlation between the normalization of immunological indicators and the good evolution of the diseases, a fact which suggests once again the existence of some immune mechanisms involved in the maintenance of sarcoidosis.
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PMID:[Preliminary results of the use of a new derivative of rifamycin SV in the treatment of sarcoidosis]. 629 6

Elevated liver enzymes are a frequent clinical problem of varying significance. In otherwise healthy individuals the most frequent causes of elevated liver enzymes are toxins such as alcohol and drugs. In this situation, further studies are usually not needed; it is sufficient to control the relevant parameters after abstinence from alcohol or withdrawal of the drug(s). In patients with known, suspected or unknown nonhepatic diseases, elevated liver enzymes can be caused by cardiovascular diseases, obesity, endocrinopathies, infectious diseases, malignancies, collagen disorders, sarcoidosis and other diseases. In this situation, sonography or liver histology frequently will be diagnostic, revealing the cause of the underlying disease as well as of the elevated liver enzymes.
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PMID:[Elevated liver enzymes of unknown etiology]. 793 57

Sarcoidosis is a chronic, multisystemic disorder of unknown etiology. The incidence of central nervous system involvement is as high as 5%. Although steroids have been the cardinal treatment for sarcoidosis, many patients become symptomatically unresponsive to them. Other patients may suffer from glucose intolerance, cataracts, and obesity, which are adverse effects of high-dose steroids. Various reports in the literature suggest that some chemotherapeutic agents and/or radiation may be useful in these situations. We present three patients with neurosarcoidosis who were treated with radiation at a single institution. We also review previous reports on radiation-treated neurosarcoid patients. While the results vary, some patients clearly derive symptomatic benefits from low-dose radiation. Since the side effects of low-dose cranial irradiation are minimal, it may be prudent to use radiation therapy for patients who are refractory to steroids or who suffer adversely from high-dose steroids.
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PMID:Radiation therapy for neurosarcoidosis: report of three cases from a single institution. 1058 Sep

The lungs are a delicate interface between the atmosphere and our bodies across which oxygen diffuses from the air we breathe to the blood which carries oxygen to the cells and mitochondria. In healthy lungs at sea level where there is a surfeit of oxygen, this process occurs easily, whereas, in lungs with disease it becomes a task which may not be fully successful and hypoxemia may ensue or worsen. At high altitude where the barometric pressure (Pb) and thus the supply of oxygen is lower, the job of getting oxygen to the blood, even in the healthy lung is more difficult, and in the diseased lung it may be impossible. This presentation will review the lungs' responses to high altitude, with emphasis on the abnormal. Both acute and chronic responses of patients with pre-existing lung disease will be reviewed. Pulmonary diseases encountered at high altitude in previously healthy people, such as high altitude pulmonary edema and chronic mountain sickness will be touched on only as they pertain to other patients. Pre-existing lung disease (with and without hypoxemia at sea level) such as obstructive lung diseases (asthma, COPD, emphysema), and restrictive lung diseases (sarcoid, asbestosis, interstitial pulmonary fibrosis) will be discussed in terms of gas exchange, lung mechanics, and treatment at high altitude. Disorders of ventilatory control; e.g., obesity-hypoventilation syndrome and sleep apnea, may present formidable problems, and guidelines for their treatment will be discussed. Infectious lung diseases; e.g., pneumonia, cystic fibrosis, and pulmonary vascular disorders such as chronic mountain sickness, primary pulmonary hypertension, and congenital absence of the pulmonary artery are important disorders that require special attention because of the accentuated hypoxic pulmonary vascular response encountered at high altitude. The purpose therefore, is to provide the medical practitioner with the insight into prevention, recognition, and treatment of pulmonary problems encountered specifically at high altitude, as well as guidance on how best to advise patients with lung disease who want to fly in airplanes and/or ascend to high altitude for work or pleasure.
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PMID:Lung disease at high altitude. 1063 92

The authors analyse hormonal and morphological characteristics of adrenal incidentalomas, i.e. pathological adrenal masses accidentally found on CT scan performed due to extraadrenal causes of other causes of adrenal pathology. The group of patients was consisted by 42 patients at the age 24-79 years (27 females and 15 males). The most frequent clinical symptoms included arterial hypertension, diabetes mellitus and obesity. CT examinations revealed 36 cases of unilateral lesions (in 21 cases the lesions were localised on the right and in 15 cases on the left) and 6 bilateral lesions. The size of adrenal masses ranged from 7 mm to 12 cm. The CT examination helped in characterising myelolipomas in 3 cases, cysts in two cases, and pre-assuming malignity in 6 cases. Hormonal analyses have revealed primary aldosteronism in 2 cases, subclinical hypercortisolism in 1, steroid enzymopathy in 2 and secondary hyperaldosteronism in 2 patients. No patient had catecholamine overproduction. 19 patients were indicated for adrenalectomy with the following histological findings.: adenoma (n = 5), cyst (n = 2), myelolipoma (n = 3), carcinoma (n = 3), feochromocytoma, ganglioneuroma, metastases, lymphoma, sarcoidosis and pseudodrenal structure--Gravitz tumor (n = 1, respectively). The size of all neoplasms exceeded 3 cm, therefore the authors recommend adrenalectomy in incidentalomas with hormonal activity exceeding 3 cm in size. (Tab. 2, Fig. 1, Ref. 17.)
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PMID:[Hormonal and morphologic characteristics of adrenal incidentalomas]. 1118 53

The use of silicone in patients with immune-compromising diseases is controversial because of fears that there may be inflammatory reactions against the material. We report the case of a patient who suffered from cutaneous sarcoidosis with bihilar lymphadenopathy and obesity (110 kg; BMI, 38 kg/m2) that was exaggerated by cortisone therapy. The patient underwent adjustable gastric banding because of aggravating comorbid hypertension and hyperlipidemia. Six months postoperatively, she had achieved an excess weight loss of 33%. During this period, she had a relapse of scar sarcoidosis of the right elbow and bilateral hilar lyphadenopathy. Although she was treated with a double dose of cortisone (8 mg daily), the scars from the laparoscopy as well as those in the gastric region adjacent to the band remained unaffected. We therefore believe that preexisting immune-compromising diseases are not an absolute contraindication against gastric banding, particularly since our patient experienced a notable improvement in her quality of life.
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PMID:Adjustable gastric banding in a patient with sarcoidosis. 1172 57

Sarcoidosis remains a fascinating illness that almost always affects the respiratory tract but often involves many other organs as well. Although many patients seem to have only an intrathoracic illness, with perhaps one other site or organ involved, others can experience a severe multi-organ disease. The inciting stimulus, even if unknown, can elicit an immunologic host response-the non-caseating granuloma-in almost every organ. It is intriguing that this stimulus can be so widespread throughout the body, while the biology of the disease can be so variable. Many series of patients with sarcoidosis have reported the multiple organs involved and the clinical presentation. Our series of 67 patients (40 female, 27 male, mean age 38.7 years +/- 13.2 (SD) at time of diagnosis) generally mirrors the clinical pattern found in five comparison series that span the past 60 years. However, more emphasis is given in this series to associated medical conditions that can complicate the presentation of sarcoidosis, as well as to co-morbid illnesses that must be managed in addition to the patient's sarcoidosis. Although most patients had intrathoracic sarcoidosis diagnosed at initial evaluation (40%), many had other organs or bodily sites involved in addition (or subsequently) as the illness evolved. Confounding the initial patient evaluation were two factors: (1) the presence of an occupational respiratory exposure(s) (n = 25 or 37% of patients); (2) a previously diagnosed malignancy (n = 6 or 9%) that heightened the possibility of a primary malignancy presenting in the chest, or the reactivation of a prior malignancy (breast, thyroid, and lymphoma) that could metastasize to the lung. Symptoms present when a patient's diagnosis was established usually differentiated respiratory and/or abdominal organ involvement. Although respiratory symptoms could be absent (n = 18 or 27%) for many patients with incidental thoracic findings, most had typical ones, including exertional dyspnea. For patients with an abdominal presenting illness (n = 11 or 16%), nonspecific digestive and abdominal symptoms were experienced as well as arthralgias. Almost every patient had at least one important other illness that factored significantly into the management of their sarcoidosis. Older patients had more illnesses, such as cardiovascular illness, diabetes mellitus, neurologic problems, and functional gastrointestinal symptoms. Depression affected all ages and was probably underrecognized; more emphasis on this illness is needed. Obesity was associated with disordered sleep syndromes, but not invariably so, as half the subjects had a good body habitus. Thus, many of the other illnesses experienced by sarcoidosis patients are common problems that middle-aged people develop. However, digestive and gastroenterological symptoms seemed disproportionately frequent in this series. This is a component of multi-organ sarcoidosis that has not received extensive coverage in the literature. Approximately one-third of sarcoidosis patients had one of two very common problems-gastroesophageal reflux or irritable bowel syndrome. But these are common problems, and it is thus necessary to separate these symptoms from those associated with abdominal visceral involvement of sarcoidosis. Although liver and/or splenic involvement with sarcoidosis do not cause organ dysfunction or insufficiency, they can contribute to abdominal symptoms. Finally, it remains of interest whether inflammatory bowel disease-Crohn's disease in particular-is another organ manifestation of sarcoidosis, or is it unrelated?
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PMID:Sarcoidosis: impact of other illnesses on the presentation and management of multi-organ disease. 1248 22


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