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At least 37 records · Page 2Linked to original sources

Non-invasive management of severe chlamydia psittaci pneumonia presenting with hypoxemia and diarrhea: a case report.

This case report describes a rare presentation of severe Chlamydia psittaci pneumonia in a 43-year-old female patient with prominent hypoxemia and gastrointestinal symptoms, and evaluates the efficacy of standardized non-invasive integrated management for critically ill patients with this atypical phenotype. The patient was admitted with lumbago, persistent high fever, progressive dyspnea, severe hypoxemia, and intractable non-bloody watery diarrhea. Chest computed tomography (CT) revealed extensive bilateral pulmonary ground-glass opacities and consolidation. Rapid and precise etiological diagnosis was achieved via targeted metagenomic next-generation sequencing (mNGS) of bronchoalveolar lavage fluid (BALF), which identified high-load Chlamydia psittaci infection, with 227,155 normalized reads and a genomic coverage of 98.6%. Comprehensive non-invasive multidisciplinary management was implemented throughout the disease course, including high-flow nasal cannula (HFNC) oxygen therapy, dual anti-infective therapy with omadacycline combined with levofloxacin, symptomatic supportive care, and standardized stepwise early rehabilitation training. Dynamic monitoring of clinical and laboratory indicators showed a gradual and sustained decline in inflammatory biomarkers (C-reactive protein,procalcitonin, interleukin-6),accompanied by progressive absorption of pulmonary lesions and recovery of respiratory function. The patient avoided invasive mechanical ventilation throughout hospitalization, was successfully weaned from HFNC on day 14 of admission, and achieved completeclinical, laboratory and radiological recovery at the 1-month follow-up. This case conforms to the CARE (CAse REports) reporting guidelines. It highlights that severe psittacosis pneumonia can present with atypical dominant manifestations of combined hypoxemia and severe gastrointestinal diarrhea, which is easily misdiagnosed clinically. Targeted mNGS enables rapid etiological confirmation of atypical severe psittacosis, and individualized non-invasive integrated management can achieve favorable prognosis in eligible critically ill patients, providing a valuable clinical reference for the standardized diagnosis and treatment of similar rare cases.

atypical clinical manifestation

Cronkhite-Canada syndrome. A case report and analytical review of 23 other cases reported in Japan.

A case study is presented of a 57-year-old male who showed typical clinical features of Cronkhite-Canada syndrome. Numerous polypoid lesions were found in the stomach, duodenum, ileum, colon and rectum accompanied with characteristic ectodermal changes. Tests indicated a protein-losing gastroenteropathy. Intestinal lactase deficiency was demonstrated by the lactose tolerance test. Scanning electronmicroscopy of the gastric and colonic mucosa revealed prominent secretion of mucoid substances and distortion in the gastric pits and colonic crypts. These abnormal findings were interpreted as having a direct relationship to the loss of protein into the gastrointestinal tract.

Adult

Idiopathic acquired sideroblastic anemia terminating in acute myelofibrosis: case report and review of leterature.

Acute myelofibrosis is a rare but distinct accelerated variant of agnogenic myeloid metaplasia that is characterized by marked anemia, peripheral blood myeloblastosis and normoblastosis, a lack of teardrop poikilocytosis, and prominent myelofibrosis. There is usually no palpable hepatosplenomegaly or lymph node enlargement. The clinical course is remarkable short. We describe a 63-year-old man who presented with idiopathic acquired sideroblastic anemia and subsequently developed acute myelofibrosis. Intensive polychemotherapy with vincristine, cytosine arabinoside, and prednisone and a later trial of oxymetholone therapy were ineffective. He died 134 days after the diagnosis of acute myelofibrosis was established. The 11 previously reported cases of acute myelofibrosis are reviewed, and the relationships of acute myelofibrosis to other myeloproliferative disorders and to idiopathic acquired sideroblastic anemia are discussed.

Acute Disease

Anaerobic liver abscess and intrahepatic metastases: a case report and review of the literature.

A patient is described in whom the first recurrence of a cloacogenic carcinoma of the rectum was an intrahepatic metastasis associated with an hepatic abscess caused by the anaerobic bacterium Peptococcus prevotii. Three previously reported cases of infection associated with hepatic tumor nodules have been found in which bacteriologic data were provided, and in all three cases anaerobic bacteria were the primary or only infection organisms. Experimental data exist which document the ability of certain anaerobic bacteria to grow selectively in tumor nodules, but not in the normal tissues of a tumor-bearing host. Since 23% of patients with liver metastases have fever and offer a clinical picture compatible with infection, occult anaerobic infection associated with liver metastases may be more common than previously recognized.

Adult

Inflammatory fibrous histiocytoma: case report.

This report concerns a patient with inflammatory fibrous histiocytoma, who in contrast to previous reported cases, has had a long survival (20 years), without evidence of recurrent disease following treatment. An interesting but nonreproducible study was the development of leukemia in 2 of 3 Swiss strain mice following the intraperitoneal injection of a saline extract of the patient's tumor.

Animals

Adenocarcinoma of the pancreas associated with hypoglycemia: case report and review of the literature.

The occurrence of profound hypoglycemia in a patient with metastatic adenocarcinoma of the pancreas is reported. In contrast to the four previously reported cases, no suggestion of excess insulin production was found. Metabolic studies in this patient suggest both increased peripheral glucose utilization and decreased hepatic glucose production as contributing factors which promoted the hypoglycemia.

Adenocarcinoma

Mesenchymal tumors associated with hypoglycemia: case report and review of the literature.

Hypoglycemia secondary to malignant tumors is rare. Mesenchymal tumors of nonpancreatic origin are the most common tumors associated with the hypoglycemia syndrome, and the clinical features of 115 reported cases are reviewed. The major anatomic distributions of the tumors are thoracic (30%) abdominal (65%), and uncommon locations (less than 5%). Approximately 50% of the tumors were resectable (59 patients), and in 60% the surgical procedure was curative. In the remaining 40% local recurrence predominated related to site of tumor and presence of contiguous organ invasion. The application of multimodality adjuvant therapy for hypoglycemia associated mesenchymal tumors should be based on an understanding of the natural history of the tumor.

Abdominal Neoplasms

Coexistent gout and rheumatoid arthritis. Case report and literature review.

A 73-year-old woman with Felty's syndrome and arthritis mutilans of long duration presented with tophaceous gout. The 7 previously reported cases of coexistent gout and rheumatoid arthritis (RA) are critically reviewed. Possible explanations for the rare coexistence of RA and gout are discussed: if uric acid is an inhibitor of the immune response, then hyperuricemia and gout could protect against development of RA. Conversely, crystalline protein binding may be a critical factor in the pathogenesis of gout, and the presence of abnormal proteins in RA could protect against gout.

Aged

Use of the carbon dioxide laser in an abdominoperineal resection for epidermoid anal carcinoma: a case report.

An abdominoperineal excision of the rectum was performed on a 52-year-old male who had epidermoid anal carcinoma. A steel scalpel was utilized for the abdominal component, but the entire perineal resection was accomplished with a carbon dioxide laser beam. We believe this to be the first reported case of an abdominoperineal resection using this new modality, which may become a useful and modern adjunct in extirpative surgery. The haemostatic effects of the laser were well demonstrated in our patient.

Anus Neoplasms

Pulmonary toxicity from carmustine (BCNU): a case report.

A patient who had a pneumonectomy for lung carcinoma was treated with carmustine when brain metastases developed. His pulmonary function was mildly compromised prior to the pneumonectomy by many years of smoking. After six months of carmustine therapy [total dose: 2,250 mg (1,200 mg/m2)] he developed interstitial pulmonary fibrosis with histologic changes consistent with drug toxicity. With seven previously reported cases of this drug-effect and the addition of our case, carmustine must be added to the list of cancer chemotherapeutic agents that can cause pulmonary toxicity.

Autopsy

Granulocytic sarcoma (chloroma) of the cerebellum and meninges a case report.

A 7-year-old boy, having had headache and vomiting for one month, was operated for a tumour in the left side of the posterior fossa. The tumour, weighing 52 g, infiltrated the dura mater, leptomeninges, and also, superficially, the left cerebellar hemisphere. The patient died five weeks after operation. Light and electron microscopical examination revealed a granulocytic sarcoma (chloroma). Pre- and postoperative blood examinations, together with postoperative and autopsy bone marrow examinations, showed no sign of acute myelogenous leukaemia. This is the second reported case of primary intracranial chloroma with no preceding sign of acute myelogenous leukemia, and the first case with cerebellar infiltration.

Cerebellar Neoplasms

Compound Heterozygous Hemoglobin Minneapolis-Laos and Codon 41/42 (-TTCT) in a Thai Female Adult: A Case Report and Literature Review.

Thalassemia is a prevalent genetic disorder in Southeast Asia. The Hemoglobin Minneapolis-Laos variant is very rarely reported with only two previously published reports that profile a total of three patients. Here, we present the first reported case of compound heterozygous β zero (β0)-thalassemia and Hemoglobin Minneapolis-Laos in a 46-year-old Thai female. She presented at Siriraj Hospital (Bangkok, Thailand) with chronic microcytic anemia, which is a more severe phenotype than would be expected from either trait alone. Initial hemoglobin electrophoresis via high-performance liquid chromatography and capillary electrophoresis revealed elevated hemoglobin A2 (5.5% and 6.3%, respectively), which is a finding consistent with a β-thalassemia trait, but this finding failed to explain the full extent of her anemia. Next-generation sequencing was then performed to investigate for a congenital red blood cell disorder. The results identified the following two mutations in the β-globin gene (HBB): heterozygous β0-thalassemia codon 41/42 (-TTCT), and HBB c.356T >A, the latter of which is consistent with hemoglobin Minneapolis-Laos. This case highlights the importance of advanced genetic testing to diagnose rare hemoglobin variants that cannot be identified by conventional investigation and further contributes to our understanding of this rare combination's clinical phenotype.

Humans

Labetalol-induced Peyronie's disease? A case report.

Peyronie's disease (induratio penis plastica) has been observed in a 58-year-old man 8 months after initiation of treatment with the new combined alpha- and beta-blocking agent, labetalol. During the last 2 months before onset of symptoms he had received 2400 mg labetalol daily. He showed no other signs of abnormal fibrous tissue production and the ANF test was negative. Cessation of the drug revealed no improvement. Peyronie's disease has also been observed in relation to treatment with propranolol, practolol and metoprolol and might be due to an impaired balance between alpha- and beta-receptors in connective tissue, but there may also be an immunological basis for the fibrosis. A possible coincidence is stressed, as the ages of the reported cases are within the range where this disease most often develops.

Ethanolamines

Double autosomal trisomy: case report (48, XX, +18, +21) and review of the literature.

A twelve-months-old female is reported with double trisomy of the autosomes 18 and 21 (48,XX,+18,+21), exhibiting the clinical features of mongolism. The findings of this patient and the data of fourteen previously reported cases with double autosomal trisomy, twelve of them mosaics, may be summarised as follows: The mean birth weight was lower than in the single trisomies D, E, and G. The distribution of the maternal ages at birth of the patients was striking: six mothers were younger than 21 years, seven mothers were older than 34 years. In those patients with prevalence of one of the two extra chromosomes in their karyotypes, the corresponding trisomy syndrome also predominated clinically. In those cases with an equal proportion of both additional chromosomes there were as many patients with clinical predominance of the one as of the other trisomy syndrome. Survival beyond the second half of the first year of life was seen only in those patients who showed the clinical picture of mongolism.

Birth Weight

Cowden's disease: a case report and review of the literature.

Cowden's disease is one of the rare genodermatoses. The most characteristic findings in this disease are cutaneous and oral lesions that are widespread papillomas and verrucous papules. On the palms the lesions are keratoses. Aside from these findings, this syndrome complex is sooner or later associated with various internal abnormalities involving the mammary, thyroid, gastrointestinal, reproductive and skeletal systems. In the 28 reported cases, 13 cancers have been found, most of the breasts. Because of the characteristic cutaneous and oral lesions, it is possible to recognize this disease early and then to follow afflicted patients closely for the possibility of development of cancers or other abnormalities of other organs.

Adult

Extrauterine mesodermal (müllerian) adenosarcoma. A case report.

Extrauterine mesodermal (müllerian) adenosarcomas have only recently been described, and this is the first reported case from Australia. These tumours fall within the category of common epithelial tumours' in the World Health Organisation (W.H.O.) classification of ovarian tumours and comprise benign looking epithelial structures (glands, papillae) in association with sarcomatous stroma. They are thus distinct from malignant mesodermal mixed tumours in which both epithelial and stromal elements are cytologically malignant.

Adult

Extrapyramidal disorder with pineal germinoma. Case report.

Space-occupying lesions of the basal ganglia are a rare cause of extrapyramidal dysfunction in children. Metastatic pineal germinoma in both basal ganglia produced dystonia in a 12-year-old boy. The literature is reviewed. Extrapyramidal manifestations in the child are compared with previously reported cases of basal ganglia neoplasms.

Basal Ganglia