Unexplained Etiology Research Articles

A Large‐scale, Rapid Public Health Response to Rabies in an Organ Recipient and the Previously Undiagnosed Organ Donor

This article describes and contrasts the public health response to two human rabies cases: one organ recipient diagnosed within days of symptom onset and the transplant donor who was diagnosed 18 months post-symptom onset. In response to an organ-transplant-related rabies case diagnosed in 2013, organ donor and recipient investigations were conducted by multiple public health agencies. Persons with potential exposure to infectious patient materials were assessed for rabies virus exposure. An exposure investigation was conducted to determine the source of the organ donor's infection. Over 100 persons from more than 20 agencies spent over 2700 h conducting contact investigations in healthcare, military and community settings. The 564 persons assessed include 417 healthcare workers [5.8% recommended for post-exposure prophylaxis (PEP)], 96 community contacts (15.6% recommended for PEP), 30 autopsy personnel (50% recommended for PEP), and 21 other persons (4.8% recommended for PEP). Donor contacts represented 188 assessed with 20.2% recommended for PEP, compared with 5.6% of 306 recipient contacts recommended for PEP. Human rabies cases result in substantial use of public health and medical resources, especially when diagnosis is delayed. Although rare, clinicians should consider rabies in cases of encephalitis of unexplained aetiology, particularly for cases that may result in organ donation.

Encephalitis-associated hospitalizations among American Indians and Alaska Natives.

Encephalitis produces considerable morbidity in the United States, but morbidity rates among American Indian/Alaska Native (AI/AN) people have not been described. Hospitalization records listing an encephalitis diagnosis were analyzed by using Indian Health Service direct/contract inpatient data. For 1998-2010, there were 436 encephalitis-associated hospitalizations among AI/AN people, an average annual age-adjusted hospitalization rate of 3.1/100,000 population. The rate for infants (11.9) was more than double that for any other age group. Death occurred for 4.1% of hospitalizations. Consistent with reports for the general U.S. population, the rate was high among infants and most (53.9%) hospitalizations were of unexplained etiology. The average annual rate during the study period appeared lower than for the general U.S. population, due particularly to lower rates in the elderly. Future community-based surveillance and mortality studies are needed to confirm these findings and examine reasons underlying the low rates of encephalitis in AI/AN people.

The role of anxiety, depression, and psychological stress on the clinical status of recurrent aphthous stomatitis and oral lichen planus

In spite of all the efforts, recurrent aphthous stomatitis (RAS) and oral lichen planus (OLP) still have unexplained etiology. The role of anxiety, depression, and psychological stress in occurrence and intensity of symptoms in RAS and OLP patients has been investigated in this study. A total of 110 patients with RAS in the acute phase and 112 patients with OLP also in acute phase participated in this study. All patients filled out questionnaires related to the primary disease (RAS/OLP) after which they took the following psychological tests: Beck Depression Inventory (BDI), The State-Trait Anxiety Inventory (STAI), and Ways of Coping Questionnaire (WCQ). According to multiple regression analysis, in patients with RAS, the highest correlation was found between results of the pain intensity and STAI test (β= 0.66; p < 0.000). In the patients with OLP, the highest correlation was found between the level of hyperkeratosis and WCQ test (β = 0.53; P < 0.000), inflammation and results of BDI test (β = 0.33; P < 0.002), and results of dynia test and STAI test (β = 0.31; P < 0.004). In this study, a high correlation between anxiety, depression, and psychological stress with symptoms of RAS and OLP has been observed.

Chylothorax in POEMS Syndrome

Chylothorax results from various causes, such as malignancy, trauma, or infection. POEMS syndrome (polyneuropathy, organomegaly, endocrinopathy, monoclonal gammopathy, and skin changes) is a multisystemic syndrome that is associated with plasma cell disorder. Pleural effusion is a common manifestation of POEMS syndrome, but the association of POEMS syndrome with chylothorax has not been reported. We report on a 61-year-old female patient who initially presented with dyspnea and bilateral leg edema. Importantly, the patient had normal renal function. Her chest X-ray and computed tomographic imaging showed bilateral pleural effusion, and her chest drainage revealed chylothorax. Detailed examination failed to reveal the definitive cause of the chylothorax. She received several treatments for chylothorax, namely, a low-fat diet or fasting, total parenteral nutrition, a somatostatin analog (octreotide), thoracic duct ligation by video-assisted thoracic surgery, and pleurodesis. However, further examination revealed endocrinopathy, monoclonal plasma cell disorder, peripheral neuropathy, and elevation of the serum level of vascular endothelial growth factor. The patient's condition was consequently diagnosed as POEMS syndrome. Eventually, her chylothorax was controlled by pleurodesis, and she was transferred to another hospital for stem cell transplantation. Herein, we report on the apparent first case of POEMS syndrome with chylothorax. In some cases of idiopathic chylothorax, the underlying primary disease may be latent, such as in the present patient. POEMS syndrome is rare, but this syndrome should be included in the differential diagnosis of chylothorax with unexplained etiology.

A child with unexplained etiology of acute pancreatitis diagnosed by endoscopic ultrasound.

A child with unexplained etiology of acute pancreatitis diagnosed by endoscopic ultrasound.

Diagnosis of Growing Pain in Bangladeshi Pediatric Population

Background: Unexplained limb pain is a major diagnostic challenge. Parents become very much worried as their physician are not sure regarding accurate diagnosis of unexplained limb pain. Majority of the limb pain are due to Growing pain which can be diagnosed by using Standard Criteria. Objectives: The purpose of the present study was to see the clinical aspects of growing pain and to determine the causes of unexplained limb pain. Methodology: This study prospectively examined the presence of growing pain in a self reported population of children with limb pain of unexplained etiology attending National Center for Control of Rheumatic Fever and Heart Disease over the period of 6 months. Results: Total 57 children of 3-12 yrs of age were enrolled in this study and out of them 43(75.4%) were diagnosed as Growing Pain. Mean age of growing pain was 7.77(2.66). 19 children (44.2%) were male and 24 (55.8%) were female. Most frequent site of pain was calf (65.1%) and 95% cases pain occur at night. Growing Pain usually occur at slow growing period (86%) than rapid growing period (14%). Conclusion: Majority of unexplained limb pain are growing pain which is benign. DOI: http://dx.doi.org/10.3329/jssmc.v5i1.16251 J Shaheed Suhrawardy Med Coll, 2013;5(1):46-48

Amniotic fluid glial fibrillary acidic protein (AF‐GFAP), a biomarker of open neural tube defects

Neural tube defects (NTDs) are usually identified by ultrasonography and confirmed by alpha-fetoprotein (AFP) assay and acetylcholinesterase (AchE) electrophoresis in amniotic fluid. Yet, both of these biomarkers can be found positive in other etiologies. Here, amniotic fluid glial fibrillary acidic protein (AF-GFAP), which was identified by a proteomic study, is shown to be a useful biomarker for NTD diagnosis. Amniotic fluid glial fibrillary acidic protein was measured by an ELISA assay in 138 cases of NTDs. Seventy samples from normal pregnancies used as controls and 27 samples giving false positive or false negative results either for AchE or AFP and corresponding to fetal death (n = 8), gastroschisis (n = 8), and unexplained etiologies (n = 11) were also tested. Whatever the gestational age, GFAP was below 0.2 ng/mL in control samples, whereas 99.1% of open NTDs (29/29 in the anterior NTD group and 80/81 in the spina bifida group) were above this threshold. Closed NTDs were all negative (28/28). None of the other samples tested were positive, except in case of fetal death (8/8). Amniotic fluid glial fibrillary acidic protein is a sensitive biomarker for open NTD diagnosis with a good negative predictive value for closed NTD. Compared with AFP and AchE, our results indicate that AF-GFAP alone is more efficient than this classical association.

Raccoon Rabies Virus Variant Transmission Through Solid Organ Transplantation

The rabies virus causes a fatal encephalitis and can be transmitted through tissue or organ transplantation. In February 2013, a kidney recipient with no reported exposures to potentially rabid animals died from rabies 18 months after transplantation. To investigate whether organ transplantation was the source of rabies virus exposure in the kidney recipient, and to evaluate for and prevent rabies in other transplant recipients from the same donor. Organ donor and all transplant recipient medical records were reviewed. Laboratory tests to detect rabies virus-specific binding antibodies, rabies virus neutralizing antibodies, and rabies virus antigens were conducted on available specimens, including serum, cerebrospinal fluid, and tissues from the donor and the recipients. Viral ribonucleic acid was extracted from tissues and amplified for nucleoprotein gene sequencing for phylogenetic comparisons. Determination of whether the donor died from undiagnosed rabies and whether other organ recipients developed rabies. In retrospect, the donor's clinical presentation (which began with vomiting and upper extremity paresthesias and progressed to fever, seizures, dysphagia, autonomic dysfunction, and brain death) was consistent with rabies. Rabies virus antigen was detected in archived autopsy brain tissue collected from the donor. The rabies viruses infecting the donor and the deceased kidney recipient were consistent with the raccoon rabies virus variant and were more than 99.9% identical across the entire N gene (1349/1350 nucleotides), thus confirming organ transplantation as the route of transmission. The 3 other organ recipients remained asymptomatic, with rabies virus neutralizing antibodies detected in their serum after completion of postexposure prophylaxis (range, 0.3-40.8 IU/mL). Unlike the 2 previous clusters of rabies virus transmission through solid organ transplantation, there was a long incubation period in the recipient who developed rabies, and survival of 3 other recipients without pretransplant rabies vaccination. Rabies should be considered in patients with acute progressive encephalitis of unexplained etiology, especially for potential organ donors. A standard evaluation of potential donors who meet screening criteria for infectious encephalitis should be considered, and risks and benefits for recipients of organs from these donors should be evaluated.

Genetic variants in the human glucocorticoid‐induced leucine zipper (GILZ) gene in fertile and infertile men

Sertoli cell only (SCO) syndrome is the predominant histology for men with non-obstructive azoospermia (NOA) and is usually of unexplained aetiology. Studies in mouse models indicated that the X-linked gene glucocorticoid-induced leucine zipper (GILZ) is essential for survival and differentiation of spermatogonia, and meiosis. GILZ deficiency results in a rapid and progressive loss of germ cells with SCO tubules and sterility in adults. The role of GILZ in human fertility has not been examined. Here we show that GILZ is localized to spermatogonia and spermatocytes in the human testis in a pattern analogous to that seen in mice. To assess the potential for an association between GILZ variants and human infertility, we sequenced the entire protein-coding regions of the GILZ gene in 65 SCO and 87 fertile Australian men. We identified six genetic variants, three of which had not been reported previously. Three variants, 107018665 G>A, 107018485 C>G and 106959283 C>T, were found at a low frequency only in SCO men. Although none of the identified variants changed the protein code, sequence analysis indicated that two variants, 107018665 G>A and 107018485 C>G, would completely abolish the exonic splicing enhancer (ESE)-binding motifs for the splicing factors SF2/ASF and SC35 respectively. This result prompted an assessment of whether these two variants were associated with male infertility in a separate population of men. We used a PCR-based SNP detection approach to screen an additional 52 NOA and 153 fertile Australian men, and 86 SCO and 54 fertile American men. None of these men carried either of these two variants. The cumulative allelic frequency of these variants is less than 1% in SCO men and no association with fertility status was observed. Our study suggests that GILZ variants are not common causes of SCO and NOA in Australian or American men.

Isolated fever induced by mesalamine treatment

Adverse reactions to mesalamine, a treatment used to induce and maintain remission in inflammatory bowel diseases, particularly ulcerative colitis, have been described in the literature as case reports. This case illustrates an unusual adverse reaction. Our patient developed an isolated fever of unexplained etiology, which was found to be related to mesalamine treatment. A 22-year-old patient diagnosed with ulcerative colitis developed a fever with rigors and anorexia 10 d after starting oral mesalamine while his colitis was clinically resolving. Testing revealed no infection. A mesalamine-induced fever was considered, and treatment was stopped, which led to spontaneous resolution of the fever. The diagnosis was confirmed by reintroducing the mesalamine. One year later, this side effect was noticed again in the same patient after he was administered topical mesalamine. This reaction to mesalamine seems to be idiosyncratic, and the mechanism that induces fever remains unclear. Fever encountered in the course of a mesalamine treatment in ulcerative colitis must be considered a mesalamine-induced fever when it cannot be explained by the disease activity, an associated extraintestinal manifestation, or an infectious etiology.

Gradual Quadriparasis: a diagnostic dilemma

This is a report of a 50 year old lady with quadriparasis who was extensively evaluated before presenting to us with imaging and nerve conduction study and diagnosed as sensory motor predominantly axonal polyneuropathy of unexplained etiology. When we evaluate the patient and a chest X-ray and further work up revealed a surprise finding.

High Gall Bladder Ejection Fraction: Whatʼs Next?

Purpose: Cholescintigraphy (HIDA scan) is a commonly used diagnostic tool in almost every hospital in the United States. A diagnosis of acalculous cholecystitis is made when the gall bladder ejection fraction (GBEF) is <35-38%. Studies have shown that low GBEF is sensitive but not a specific indicator of gall bladder inflammation. The higher spectrum of gall bladder ejection fraction has largely remained unnoticed as it is considered normal. However our perspective changed after we encountered this particular case. A 35-year-old woman was seen for chronic abdominal pain of unexplained etiology over the past year. Her past medical history included prolonged course of Clostridium difficile infection which was treated. She also had lupus anticoagulants. The patient reported persistent right upper quadrant discomfort with paroxysms of severe exacerbation which sometimes even woke her from sleep. The pain worsened postprandially. She had constant nausea without vomiting. She also had episodic watery diarrhea about 2 or 3 days out of the week. There was no rectal bleeding. Her weight had been stable. The pain was severe enough to interfere with her daily life and with her work at times. A right upper quadrant sonogram was performed and did not demonstrate any gallstones. Following this an extensive workup was done including upper endoscopy, colonoscopy, capsule endoscopy, small bowel series, CAT scan, and mesenteric angiography. All showed no significant findings. A functional HIDA scan was performed with CCK Administration (0.02 μg/kg over 60 minutes), and her gallbladder ejection fraction was noted to be 94%. This was the only significant finding in her workup. Possible etiologies at this time were endometriosis or gallbladder dysfunction. The patient underwent a planned exploratory laparoscopy and cholecystectomy. The histopathology showed chronic cholecystitis. The patient had partial improvement in symptoms for a short duration; however her symptoms recurred soon after. Treatment options for patients with high gall-bladder EF (>80%) that have unremitting symptoms remains obscure. There are no major studies or guidelines on how to approach patients with hyperdynamic gall bladders. This case illustrates that high EF can be associated with inflammation. It is unfortunate that this particular patient did not get better, but small numbers of studies have shown benefit from surgery. We need an answer to “What's next?” when patients have high gallbladder ejection fractions.

A High Dose Intravenous Immunoglobulin Therapy for Women with Four or More Recurrent Spontaneous Abortions

Recurrent spontaneous abortion (RSA) may have immunological etiology. The aim of this study was to assess the efficacy of a high dose intravenous immunoglobulin (HIVIg) therapy, in which 20 g of intact type immunoglobulin was infused daily for 5 days during early gestation, for women who had a history of four or more consecutive spontaneous abortions of unexplained etiology. A total of 60 pregnant RSA women underwent HIVIg therapy, and the pregnancy outcome was assessed. The live birth rate was 73.3% (44/60). Fifteen pregnancies ended in spontaneous abortion, and one ended in intrauterine fetal death. In 11 of the 15 spontaneous abortions, fetuses had abnormal chromosome karyotype. When the 11 pregnancies with abnormal chromosome karyotype were excluded, the live birth rate was as high as 89.8% (44/49). The HIVIg therapy may be effective for severe cases of unexplained RSA.

Interrogating Altered DNA Methylation in Human Endometriotic Stromal Cells.

Endometriosis is a benign gynecological disease causing infertility, adhesions and chronic pain in approximately 10% of reproductively-aged women due to the extrauterine growth of endometrial stromal and glandular cells. Endometriotic lesions resemble the eutopic endometrium both morphologically and in their response to estrogen, which potently stimulates stromal cell proliferation and gene expression. However, diseased cells are characterized by a number of cellular and molecular defects with unexplained etiology. Growing evidence suggests that abnormal DNA methylation is an underlying problem in the disease, since such epigenetic changes can be both mutable and heritable. Aberrant DNA methylation within the promoters of estrogen receptor beta, steroidogenic factor 1, aromatase, and HOXA10—all genes tied directly to the pathology of endometriosis—is now known to alter their expression in endometriotic cells. We hypothesize that deviations in DNA methylation may accrue in the stromal cells of endometriotic lesions, fundamentally altering patterns of gene expression and changing the response of these cells to steroid hormone signaling during the menstrual cycle. We used normal and endometriotic stromal cells to identify differences in genome-wide methylation and gene expression using Illumina's Infinium HumanMethylation450 and HumanHT-12v4 Expression BeadChips. Normal endometrial stromal cells (HESC) were obtained from six patients without endometriosis undergoing hysterectomy for benign complications. Endometriotic stromal cells (OSIS) were obtained from six patients having surgical removal of ovarian endometriotic cysts. All patients were premenopausal, and underwent surgery during the proliferative phase of their menstrual cycle having received no preoperative hormonal therapy. The gene expression results identified 982 differentially regulated genes between the two groups (515 upregulated genes, and 467 downregulated). The methylation array revealed 53,977 cytosine-guanine sites (CpGs) that were differentially methylated between HESC and OSIS (beta>0.1; fold change>1.5; p<0.05). Only 11% of these differences were observed in canonical CpG islands, whereas more than 55% were located in regions unassociated with any CpG island. Notably, 3817 of the differentially methylated CpGs were mapped to genes also showing altered expression between the two groups. Within this group, 2502 CpGs were hypermethylated in OSIS, and methylation was inversely correlated with gene expression. Furthermore, gene ontology analysis of the genes that were hypermethylated in OSIS identified an enrichment of biological process involved in urogenital and female sexual development, whereas hypomethylated genes were enriched in biological processes regulating cell migration and locomotion. We have uncovered global differences in methylation between normal and diseased cells that correlate with extensive changes in gene expression, and we predict that these epigenetic changes are involved in the establishment and progression of endometriosis.

Unexplained neonatal jaundice as an early diagnostic sign of urinary tract infection

Hyperbilirubinemia is one of the presenting signs of bacterial infection in newborns, and the association of neonatal jaundice with urinary tract infection (UTI) has been particularly emphasized. The aim of this study was to determine the prevalence of UTI in asymptomatic jaundiced neonates younger than 4 weeks old. We prospectively evaluated 120 asymptomatic jaundiced and 122 healthy neonates without jaundice younger than 4 weeks old for UTI. Patients with UTI, defined as >10,000 colony-forming units of a single pathogen per milliliter urine obtained by bladder catheterization, were evaluated for sepsis. Of 120 asymptomatic jaundiced neonates with a mean age of 7 ± 4 days, 15 (12.5%) had a UTI. Of 122 healthy neonates, positive urine cultures from a urine bag were found in eight cases; however on reevaluation, urine cultures from bladder catheterization were negative. The most common pathogen isolated from the UTI cases was Klebsiella pneumoniae. Also, unconjugated hyperbilirubinemia was detected in all jaundiced patients with UTI. UTI was found in 12.5% of the asymptomatic jaundiced neonates with the onset of unconjugated hyperbilirubinemia in the first week of life. Therefore, we suggest that urine culture should be considered as a part of the diagnostic evaluation of jaundiced neonates older than 3 days with an unexplained etiology.

From suspicion of liver metastases to the diagnosis of Wilson disease in a patient with seminoma

Wilson disease is an autosomal recessive disorder characterized by copper accumulation in the liver, brain, kidneys, and cornea due to inadequate biliary copper excretion. It should be considered especially in young patients who have findings of liver disease with unexplained etiology. Clinical presentation of the disease can be variable, and different types of parenchymal changes of the liver can be seen on imaging modalities. Multiple nodular lesions mimicking metastases can be detected. This condition can obligate physicians to screen for a malignant disease. Moreover, it may cause misdiagnosis as advanced stage of disease when coexistent with a malignancy. The coexistence of Wilson disease with some malignant diseases has been reported; however, coexistence with seminoma was not reported before. Approximately 40% of testicular cancers are pure seminoma. Liver metastases are rare in seminoma. In this article, a case of Wilson cirrhosis is reported. The patient was first followed with diagnosis of seminoma with suspicion of liver metastases.

What do we know about chronic kidney disease in India: first report of the Indian CKD registry

BackgroundThere are no national data on the magnitude and pattern of chronic kidney disease (CKD) in India. The Indian CKD Registry documents the demographics, etiological spectrum, practice patterns, variations and special characteristics.MethodsData was collected for this cross-sectional study in a standardized format according to predetermined criteria. Of the 52,273 adult patients, 35.5%, 27.9%, 25.6% and 11% patients came from South, North, West and East zones respectively.ResultsThe mean age was 50.1 ± 14.6 years, with M:F ratio of 70:30. Patients from North Zone were younger and those from the East Zone older. Diabetic nephropathy was the commonest cause (31%), followed by CKD of undetermined etiology (16%), chronic glomerulonephritis (14%) and hypertensive nephrosclerosis (13%). About 48% cases presented in Stage V; they were younger than those in Stages III-IV. Diabetic nephropathy patients were older, more likely to present in earlier stages of CKD and had a higher frequency of males; whereas those with CKD of unexplained etiology were younger, had more females and more frequently presented in Stage V. Patients in lower income groups had more advanced CKD at presentation. Patients presenting to public sector hospitals were poorer, younger, and more frequently had CKD of unknown etiology.ConclusionsThis report confirms the emergence of diabetic nephropathy as the pre-eminent cause in India. Patients with CKD of unknown etiology are younger, poorer and more likely to present with advanced CKD. There were some geographic variations.

Romatoid Artrite Sekonder Difüz Büyük B Hücreli Lenfoma

Lymphoid malignancies are a heterogeneous group of neoplasms originating from B- and T-lymphocytes with an unexplained etiology. Several autoimmune conditions, especially Sjögren syndrome and rheumatoid arthritis (RA) are associated with lymphoma. Immunosuppressive agents like methotrexate may cause Epstein-Barr virus reactivation that can lead to development of lymphoma. The risk of lymphoma, especially Hodgkin lymphoma, is increased up to 2-3 times in RA patients compared to normal population. The cause of increased risk is independent from the immunosuppressive medication. We report here an unusual case of diffuse large B-cell lymphoma in a patient with RA receiving methotrexate treatment, after obtaining the approval of the informed consent of the patient.

Implication of Protein-C in Thrombophilic State and Metastatic Dissemination

The coagulation/fibrinolytic system controls the intravascular fibrin homeostasis; in addition to participating in a wide variety of physio-pathological processes. The components of the system have an influence on tumor metastasis, growth and invasion. This is a result of their involvement in tumor matrix construction, angiogenesis and cell migration. Thrombosis of unexplained etiology among healthy and cancer patients; is a major cause of death. Several homeostatic markers are currently used to predict the advent of thrombosis. However, none of these markers directly indicate the course and progression of the disease; thus thrombosis remains unexplained. Endothelial Protein C Receptor EPCR gene carries 13 single nucleotide polymorphisms, which define 3 haplotypes: A1, A2 and A3. One of these, A3, encodes a protein, which is more sensitive than the other two in shedding enzymes. High levels of protein C are determined by PROCR haplotype 3. A3 haplotype reflects a high soluble Endothelial Protein C Receptor (sEPCR) level. Therfore, it is a candidate risk factor for venous thrombosis. We observed that the plasma concentration of sEPCR in cancer patients was much higher than that observed in controls. We suggest that sEPCR released from malignant cells could serve as a “trap” for protein C, preventing it&apos;s binding to EPCR on the surface of endothelial cells and induced thrombotic state. We developed a method, based on activated Partial Thromboplastin Time, in order to analyze the ability of (EPCR) on the cancer cell membrane to trap circulating Activated Protein C (APC). This test is in conjunction with other specific tests used for assessing thrombotic state, such as the one to quantitate soluble fibrin and d-dimer. Previous study of lung cancer patients, done by Department of Pathology, Free University Medical Center 2002, revealed a marked association between high EPCR levels; and poor survival or relapse in patients with stage I lung adenocarcinoma. The aim of our study is to investigate the role of sEPCR as a cause of thrombotic disorder, among cancer patients. Furthermore, to detect whether EPCR of cancer cells is haplotype 3, that affect on the level of sEPCR.

Unexplained acute respiratory distress syndrome after varicocele surgery; report of a case

Acute respiratory distress syndrome (ARDS) is a syndrome of acute respiratory failure that is associated with several clinical disorders including direct pulmonary injury and indirect pulmonary injury. Vascular surgical patients are at risk for ARDS. A 23-year old man was admitted to our respiratory intensive care unit with short of breath, cyanosis and agitation which was developed acutely after varicocele operation under epidural anesthesia lasted for one hour. Bupivacain, fentanyl, remifentadyl and midazolam were used for epidural anesthesia. Oxygen saturation was 81% while breathing oxygen at FiO(2) of 0.5. Arterial blood gas analysis showed hypoxemia with respiratory alkalosis with a PaO(2)/FiO(2) score of 100. A chest radiograph demonstrated bilateral alveolar opacities. Bedside echocardiography was performed which revealed no evidence of global hypokinesia, all the chambers were normal, left ventricular ejection fraction was 65%. A diagnosis of ARDS was made and the patient was initiated on noninvasive mechanical ventilation. He was gradually weaned off the NIMV and discharged after a total hospital stay of four days. We report an ARDS case due to unexplained etiology after varicocele operation under epidural anesthesia and managed successfully with noninvasive mechanical ventilation.