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1.  Histoplasmosis Presenting as Granulomatous Hepatitis: Case Report and Review of the Literature 
Case Reports in Medicine  2014;2014:879535.
Background. Histoplasma capsulatum is the most common endemic mycosis in the United States and is a frequent cause of opportunistic infection in immunodeficient hosts. Histoplasmosis is most often self-limiting and goes unrecognized in the immunocompetent population but can progress to disseminated histoplasmosis in patients with an impaired immune system. Liver involvement as a part of disseminated histoplasmosis which usually originates in the lung is well known. However, extrapulmonary hepatic histoplasmosis as a primary manifestation is extremely rare. Case Presentation. We report a rare case of histoplasmosis that presented as persistent fever and abnormal liver function tests in a 66-year-old female with rheumatoid arthritis, receiving infliximab. Conclusion. Emphasizing histoplasmosis as a major cause of acute granulomatous hepatitis and fever of unknown origin in cell mediated immunodeficient population, this case highlights the need for high index of suspicion and the importance of prompt diagnosis since any delay of treatment can be life threatening in this population.
PMCID: PMC4070324
2.  State of Oral Mucosa as an Additional Symptom in the Course of Primary Amyloidosis and Multiple Myeloma Disease 
Case Reports in Medicine  2014;2014:293063.
Multiple myeloma (myeloma multiplex (MM)) is a malignant non-Hodgkin's lymphoma derived from B cell. Its essence is a malignant clone of plasma cells synthesizing growth of monoclonal immunoglobulin, which infiltrate the bone marrow, destroy the bone structure, and prevent the proper production of blood cells components. The paper presents a case of 62-year-old patient who developed symptoms in addition to neurological and haematological changes in the oral mucosa in the course of multiple myeloma. The treatment resulted in partial improvement. The authors wish to draw attention not only to nonspecificity and rarity of changes in the mouth which can meet the dentist but also to the complexity of the multidisciplinary therapy patients diagnosed with MM.
PMCID: PMC4070359
3.  Inefficacy or Paradoxical Effect? Uveitis in Ankylosing Spondylitis Treated with Etanercept 
Case Reports in Medicine  2014;2014:471319.
Ankylosing spondylitis (AS) is presented with axial and peripheral articular involvement. Uveitis is a severe and rather specific manifestation of AS. Biologics targeting tumor necrosis factor (TNF) α are effective on both articular and ocular manifestations of disease. The occurrence of uveitis in patients that never had eye involvement or the relapse of uveitis is described during anti-TNFα treatment. The frequency of these events is slightly higher during therapy with etanercept. The available TNFα blockers show different pharmacokinetics and pharmacodynamics yielding different biological effects. There is an ongoing debate whether uveitis during anti-TNFα has to be considered as paradoxical effect or an inadequate response to therapy. Here, we present a case report and review what the evidences for the two hypotheses are.
PMCID: PMC4065678  PMID: 24991219
4.  Rare Manifestation of a Rare Disease, Acute Liver Failure in Adult Onset Still's Disease: Dramatic Response to Methylprednisolone Pulse Therapy—A Case Report and Review 
Case Reports in Medicine  2014;2014:375035.
Adult onset Still's disease (AOSD) is a rare systemic inflammatory disorder of unknown etiology. It is characterized by daily fevers, arthralgias or arthritis, typical skin rash, and leukocytosis. Hepatic involvement is frequently observed in the course of AOSD with mildly elevated transaminases and/or hepatomegaly. Fulminant hepatic failure, occasionally requiring urgent liver transplantation, is a rare manifestation of AOSD. Here, we present a case of 22-year-old woman with no significant medical history who initially came with fever, arthralgias, myalgias, generalized weakness, and sore throat. Laboratory data showed mildly elevated transaminases and markedly elevated ferritin levels. She was diagnosed with AOSD based on Yamaguchi diagnostic criteria and was started on prednisone. Three months later, while she was on tapering dose of steroid, she presented with fever, abdominal pain, jaundice, and markedly elevated transaminases. Extensive workup excluded all potential causes of liver failure. She was diagnosed with AOSD associated acute liver failure (ALF). Intravenous (IV) methylprednisolone pulse therapy was started, with dramatic improvement in liver function. Our case demonstrated that ALF can present as a complication of AOSD and IV mega dose pulse methylprednisolone therapy can be employed as a first-line treatment in AOSD associated ALF with favorable outcome.
PMCID: PMC4065746  PMID: 24991218
5.  Graves' Disease and Treatment Effects on Warfarin Anticoagulation 
Case Reports in Medicine  2014;2014:292468.
Background. Hyperthyroidism causes an increased hypoprothrombinemic response to warfarin anticoagulation. Previous studies have demonstrated that patients with hyperthyroidism require lower dosages of warfarin to achieve a therapeutic effect. As hyperthyroidism is treated and euthyroidism is approached, patients may require increasing warfarin dosages to maintain appropriate anticoagulation. We describe a patient's varying response to warfarin during treatment of Graves' disease. Case Presentation. A 48-year-old African American female presented to the emergency room with tachycardia, new onset bilateral lower extremity edema, gradual weight loss, palpable goiter, and generalized sweating over the prior 4 months. She was admitted with Graves' disease and new onset atrial fibrillation. Primary stroke prophylaxis was started using warfarin; the patient developed a markedly supratherapeutic INR likely due to hyperthyroidism. After starting methimazole, her free thyroxine approached euthyroid levels and the INR became subtherapeutic. She remained subtherapeutic over several months despite steadily increasing dosages of warfarin. Immediately following thyroid radioablation and discontinuation of methimazole, the patient's warfarin dose and INR stabilized. Conclusion. Clinicians should expect an increased response to warfarin in patients with hyperthyroidism and close monitoring of the INR is imperative to prevent adverse effects. As patients approach euthyroidism, insufficient anticoagulation is likely without vigilant follow-up, INR monitoring, and increasing warfarin dosages.
PMCID: PMC4065757  PMID: 24991217
6.  Clinical Case of Immune Dysregulation, Polyendocrinopaty, Enteropathy, X-Linked (IPEX) Syndrome with Severe Immune Deficiency and Late Onset of Endocrinopathy and Enteropathy 
Case Reports in Medicine  2014;2014:564926.
Objective. To describe the clinical characteristics of IPEX syndrome in a child with FOXP3 mutation. Clinical Case. A boy aged 2.3 years was born from first normal pregnancy with a weight of 3420 gr. Family History. Two brothers of the mother died before the age of 3 years with severe infections, diarrhea, erythroderma, and elevated immunoglobulins class E (IgEs). Since first month of life, our patient suffered from septicemia, pneumonias, pyelonephritis, and meningitis, accompanied with eczematous dermatitis and IgEs up to 4000 IU/L (normal <10). At the age of 1.6 years, he developed type 1 diabetes mellitus (T1DM). He was underweighted (−3.42 SDS) and had some phenotypic features like coarse face, muscle hypotonia, joint hyperextensibility, eczematous dermatitis, and subcutaneous cold abscesses. Autoimmune thyroiditis and celiac disease were excluded. After diabetes, intermittent watery diarrhea appeared with progression to severe intractable form. Finally, aggravating symptoms of nephritis, cachexia, and respiratory insufficiency were the cause for his death at the age of 2 years and 3 months. The DNA analysis at the University of Exeter Medical School established mutation at exon 10 of FOXP3 gene c.1010G >A, p. (Arg337Gln), which confirmed IPEX syndrome. The same mutation in heterozygotic state was found in the mother. A prenatal diagnosis of her second pregnancy ensured a daughter carrier of the mutation.
PMCID: PMC4058591  PMID: 24982679
7.  Solid Pseudopapillary Neoplasms of the Pancreas: A Report of Two Cases 
Case Reports in Medicine  2014;2014:356379.
Solid pseudopapillary neoplasms of the pancreas are uncommon, accounting for only 1-2% of all pancreatic neoplasms. These tumors are being detected at an increased rate, probably due to the increased awareness and the liberal use of imaging. We report two cases of patients with solid pseudopapillary pancreatic tumors and review the existing literature.
PMCID: PMC4058598  PMID: 24982677
8.  Multimodal Surgical and Medical Treatment for Extensive Rhinocerebral Mucormycosis in an Elderly Diabetic Patient: A Case Report and Literature Review 
Case Reports in Medicine  2014;2014:527062.
Diabetes is a well-known risk factor for invasive mucormycosis with rhinocerebral involvement. Acute necrosis of the maxilla is seldom seen and extensive facial bone involvement is rare in patients with rhino-orbital-cerebral mucormycosis. An aggressive surgical approach combined with antifungal therapy is usually necessary. In this report, we describe the successful, personalized medical and surgical management of extensive periorbital mucormycosis in an elderly diabetic, HIV-negative woman. Mono- or combination therapy with liposomal amphotericin B (L-AmB) and posaconazole (PSO) and withheld debridement is discussed. The role of aesthetic plastic surgery to preserve the patient's physical appearance is also reported. Any diabetic patient with sinonasal disease, regardless of their degree of metabolic control, is a candidate for prompt evaluation to rule out mucormycosis. Therapeutic and surgical strategies and adjunctive treatments are essential for successful disease management. These interventions may include combination therapy. Finally, a judicious multimodal treatment approach can improve appearance and optimize outcome in elderly patients.
PMCID: PMC4058497  PMID: 24982678
9.  Hemophagocytic Syndrome Associated with Immune Reconstitution Inflammatory Syndrome in a Patient with AIDS Related Burkitt's Leukemia/Lymphoma 
Case Reports in Medicine  2014;2014:308081.
Highly active antiretroviral therapy (HAART) has markedly decreased human immunodeficiency virus- (HIV-) related mortality and the incidence of opportunistic infections. The dramatic reduction in HIV-1 RNA and increase in CD4 lymphocyte count mean a recovery in immune function. This restoration in immune function may be associated with paradoxical deterioration in subclinical opportunistic infections in some patients, a condition called immune reconstitution inflammatory syndrome (IRIS). IRIS, a “paradoxical” inflammatory response to either previously treated or subclinical infections or noninfectious diseases, can manifest during the restoration phase of immunity hemophagocytic syndrome (HS) which is a very rare complication in patients with acquired immune deficiency syndrome (AIDS). We describe a case of hemophagocytic syndrome associated with IRIS in a patient with AIDS related Burkitt's leukemia/lymphoma (BL). IRIS was probably the cause of hemophagocytosis for our patient. Zoster infection may facilitate to IRIS. With the increasing number of people with HIV infection and the accompanying use of HAART, much more clinical manifestations of IRIS will be experienced especially in patients given high dose chemotherapy, just like in our case.
PMCID: PMC4058513  PMID: 24987420
10.  Reversible Stress Cardiomyopathy Presenting as Acute Coronary Syndrome with Elevated Troponin in the Absence of Regional Wall Motion Abnormalities: A Forme Fruste of Stress Cardiomyopathy? 
Case Reports in Medicine  2014;2014:796202.
We present a case of reversible stress cardiomyopathy in a surgical patient, described here as a forme fruste due to its atypical features. It is important to recognize such unusual presentation of stress cardiomyopathy that mimics acute coronary syndrome. Stress cardiomyopathy commonly presents as acute coronary syndrome and is characterized by typical or atypical variants of regional wall motion abnormalities. We report a 60-year-old Caucasian male with reversible stress cardiomyopathy following a sternal fracture fixation. Although the patient had several typical features of stress cardiomyopathy including physical stress, ST-segment elevation, elevated cardiac biomarkers and normal epicardial coronaries, there were few features that were atypical, including unusual age, gender, absence of regional wall motion abnormalities, high lateral ST elevation, and high troponin-ejection fraction product. In conclusion, this could represent a forme fruste of stress cardiomyopathy.
PMCID: PMC4058213  PMID: 24976829
11.  Endovascular Treatment of Vertebral Column Metastases Using Intra-Arterial Cisplatin: Pilot Experience 
Case Reports in Medicine  2014;2014:915904.
Background and Importance. Treatment of spinal column metastatic tumors is challenging, especially in the setting of progressive disease despite previous radiation and chemotherapy. Intra-arterial chemotherapy is an uncommonly used but established treatment for head and neck cancers, retinoblastoma, and glioblastoma. The author reports extension of the IAC concept to vertebral metastatic tumors. Clinical Presentation. Two patients with intractable spinal pain secondary to spinal metastatic involvement at T11-L1 segments were treated with intra-arterial injections of cisplatin, with simultaneous sodium thiosulfate chelation. The first patient, a 60-year old female with metastatic lung carcinoma underwent, three cycles of therapy over a 9-week period; the treated regions demonstrated bone remodeling and sclerosis. The second case was a 40-year old male with malignant pheochromocytoma, who underwent a single treatment and succumbed 5 weeks later from progressive widespread disease. Both patients reported significant pain relief and neither of them exhibited a decline in neurologic function. Conclusion. The intra-arterial delivery of cisplatin appeared to be well tolerated in the two cases. In the case with the longest survival, the treated vertebral segments became more sclerotic, consistent with biomechanical stabilization. Endovascular treatment of spinal metastases may hold promise, especially as newer categories of biologic agents become more widely available.
PMCID: PMC4055045  PMID: 24963303
12.  A New Case of Syringocystadenocarcinoma Papilliferum: A Rare Pathology for a Wide-Ranging Comprehension 
Case Reports in Medicine  2014;2014:453874.
We report a new case of p63/cytokeratin 7 (CK7) positive syringocystadenocarcinoma papilliferum (SCACP), on the shoulder of an 88-year-old man, with superficial dermal infiltration and squamoid differentiation. We describe the 24th case of SCACP, the malignant counterpart of syringocystadenoma papilliferum (SCAP). At the present, we do not know whether SCACP arises from eccrine or apocrine glands because of the contrasting opinions in the literature. Only few histochemical and ultrastructural studies have previously advised that SCACP could arise from pluripotent stem cells. Through our case, we wish to suggest the stem cell-like properties of the syringocystadenocarcinoma papilliferum. This rare neoplasm shows two different patterns of stem cell marker expression in the glandular and squamous components, respectively. For the double phenotype of SCACP, we propose it like an intriguing model to study histogenesis and stem cell properties for more wide-ranging epithelial tumors.
PMCID: PMC4052556  PMID: 24959179
13.  Evaluation of a Three-Stage Method for Improving Mandibular Retrognathia with Labially Inclined Incisors Using Genioplasty, Segmental Osteotomy, and Two-Jaw Surgery 
Case Reports in Medicine  2014;2014:314179.
We have sometimes encountered difficulty in improving labially inclined teeth, particularly in patients with mandibular retrognathia, because the symphysis menti is often thin and insufficient space is available to permit sagittal rotation of the teeth without root exposure from the alveolar bone. We have previously described a three-stage method to overcome this problem, involving genioplasty for improving the retruded chin, and to construct the infrastructure for subsequent subapical segmental alveolar osteotomy, subapical segmental alveolar osteotomy itself, and, finally, two-jaw surgery. Bone augmentation with thin cortical bone at the gap created on the upper surface of the advanced genial segment was also addressed in the previous report. In the present study, to confirm the benefits of the three-stage method using objective data, cephalometric evaluation was performed in each step. In all cases, pogonion (Pog) was moved forward substantially. Net linear forward movement of Pog and net changes in SN-Pog were from 12 mm to 20 mm and from 4.8° to 7.0°, respectively. Angle of mandibular incisors and interincisal angle also improved to desirable levels. Although this method requires three separate surgeries, the approach safely improves the clinical situation and accentuates treatment efficacy.
PMCID: PMC4053231  PMID: 24963302
14.  Pulp Revascularization in Immature Permanent Tooth with Apical Periodontitis Using Mineral Trioxide Aggregate 
Case Reports in Medicine  2014;2014:564908.
Mineral trioxide aggregate (MTA) is a material that has been used worldwide in several clinical applications, such as apical barriers in teeth with immature apices, repair of root perforations, root-end filling, pulp capping, and pulpotomy. The purpose of this case report was to describe successful revascularization treatment of an immature mandibular right second premolar with apical periodontitis in a 9-year-old female patient. After preparing an access cavity without anesthesia, the tooth was isolated using a rubber dam and accessed. The canal was gently debrided using 5% sodium hypochlorite (NaOCl) and 3% hydrogen peroxide irrigant. And then MTA was packed into the canal. X-ray photographic examination showed the dentin bridge 5 months after the revascularization procedure. Thickening of the canal wall and complete apical closure were confirmed 10 months after the treatment. In this case, MTA showed clinical and radiographic success at revascularization treatment in immature permanent tooth. The successful outcome of this case suggests that MTA is reliable and effective for endodontic treatment in the pediatric dentistry.
PMCID: PMC4052928  PMID: 24959181
15.  Pulmonary Phaeohyphomycosis Caused by Phaeoacremonium in a Kidney Transplant Recipient: Successful Treatment with Posaconazole 
Case Reports in Medicine  2014;2014:902818.
We report a rare case of pulmonary phaeohyphomycosis in a 49-year-old woman 6 years after kidney transplantation. She presented with dyspnea, cough, and fatigue. Her chest CT scan revealed nodular opacities in the right upper lung. A fine needle aspirate biopsy culture yielded Phaeoacremonium and surgical pathology of the biopsy showed chronic inflammation. We successfully treated her with posaconazole and managed drug interactions between posaconazole and tacrolimus. This is the second reported case of biopsy-proven pulmonary infection by Phaeoacremonium in a kidney transplant recipient and successfully treated with posaconazole.
PMCID: PMC4053290  PMID: 24959182
16.  Persistent Salmon Patch on the Forehead and Glabellum in a Chinese Adult 
Case Reports in Medicine  2014;2014:139174.
Salmon patches are present in approximately 44% of all neonates. The lesions tend to fade with time and those on the glabellum, eyelids, nose, and upper lip are rarely detected after the age of 6. We report a 33-year-old Chinese female with a salmon patch on the forehead and glabellum. To our knowledge, the occurrence of a salmon patch on the forehead and glabellum in adulthood has not been reported. The persistent salmon patch on the face of an adult is benign and not associated with any neurocutaneous syndrome or underlying vascular abnormality. The color of the lesion can be ameliorated with laser therapy if cosmesis is a concern.
PMCID: PMC4053299  PMID: 24963301
17.  Reporting a Rare Case of Pleomorphic Adenoma of the Breast 
Case Reports in Medicine  2014;2014:387183.
Pleomorphic adenoma (PA) is the most common tumor type in the salivary gland. PA is uncommon in the breast tissue. Only 73 cases of PA of the breast have been reported in the world literature. We are reporting the 74th case of PA of the breast. A 61-year-old woman was referred to Shahid Beheshti Hospital Obstetric Clinic with bloody painless discharge from the right nipple. A bean size mass was detected immediately below the right nipple. After an excisional biopsy, the pathologist found proliferation in epithelial and myoepithelial cells that had small and multiple nuclei, myxoid and chondroid stroma. Immunohistochemistry stain was positive for S-100 and patchy for GFAP in tumor cells and for SMA around the tubule-glandular and tumor cell aggregates and suggested PA of the breast. It is essential for the pathologists to consider PA of the breast as a differential diagnosis of a rounded circumscribed mass in the juxta-areolar areas. Careful paraffin sections should be performed to avoid an unnecessary mastectomy.
PMCID: PMC4036713  PMID: 24899901
18.  An Interesting Case of Life-Threatening Hypercalcemia Secondary to Atypical Parathyroid Adenoma versus Parathyroid Carcinoma 
Case Reports in Medicine  2014;2014:473814.
Context. Severe hypercalcemia is a life-threatening condition. Atypical parathyroid adenoma and parathyroid carcinomas are uncommon causes which can be difficult to differentiate. Objective. We report a case of a 36-year-old male with very high serum calcium due to a possible atypical parathyroid adenoma versus parathyroid carcinoma. Case Illustration. A serum calcium level of 23.2 mg/dl was noted on admission. He was initially treated with IV hydration, pamidronate, and salmon calcitonin to lower his calcium levels. He also underwent a surgical en bloc resection of parathyroid mass. Pathology showed a mixed picture consistent with possible atypical adenoma versus parathyroid carcinoma. However, due to the possible involvement of the recurrent laryngeal nerve, parathyroid carcinoma was more likely. Also after operation the patient developed hungry bones syndrome and his calcium was replaced vigorously. He continues to be on calcium, vitamin D, and calcitriol supplementation. Results. A review of the literature was conducted to identify previous studies pertaining to parathyroid adenomas and parathyroid cancer. Conclusion. We thereby conclude that hypercalcemia requires very careful monitoring especially after operation. Also it can be very difficult to distinguish between atypical parathyroid adenomas and parathyroid carcinomas as in our case and no clear cut guidelines yet exist to differentiate the two based on histology.
PMCID: PMC4053223  PMID: 24959180
19.  Esophageal Rupture as a Primary Manifestation in Eosinophilic Esophagitis 
Case Reports in Medicine  2014;2014:673189.
Eosinophilic esophagitis (EoE) is a chronic inflammatory process characterized by symptoms of esophageal dysfunction and, histologically, by eosinophilic infiltration of the esophagus. In adults, it commonly presents with dysphagia, food impaction, and chest or abdominal pain. Chronic inflammation can lead to diffuse narrowing of the esophageal lumen which may cause food impaction. Endoscopic procedures to relieve food impaction may lead to complications such as esophageal perforation due to the friability of the esophageal mucosa. Spontaneous transmural esophageal rupture, also known as Boerhaave's syndrome, as a primary manifestation of EoE is rare. In this paper, we present two adult patients who presented with esophageal perforation as the initial manifestation of EoE. This rare complication of EoE has been documented in 13 other reports (11 adults, 2 children) and only 1 of the patients had been previously diagnosed with EoE. A history of dysphagia was present in 1 of our patients and in the majority of previously documented patients. Esophageal perforation is a potentially severe complication of EoE. Patients with a history of dysphagia and patients with spontaneous esophageal perforation should warrant an evaluation for EoE.
PMCID: PMC4037595  PMID: 24899902
20.  Photodynamic Therapy for Diffuse Choroidal Hemangioma in Sturge-Weber Syndrome 
Case Reports in Medicine  2014;2014:452372.
Purpose. To report the treatment outcome of photodynamic therapy with verteporfin (PDT) for exudative retinal detachment (RD) associated with diffuse choroidal hemangioma in Sturge-Weber syndrome (SWS). Methods. An interventional case report of a 10-year-old girl with SWS who developed an exudative RD (visual acuity hand motions) that was treated with PDT. She was treated with a first session of multispot PDT. Posteriorly, a choroidotomy for drainage of subretinal fluid was created, combined with an intravitreal injection of gas (SF6) and cryoapplication. Finally, a second session of PDT was applied. Results. Subretinal fluid resolved over a period of one year and visual acuity increased to 20/125. Conclusions. PDT is an effective therapeutic option for exudative RD associated with diffuse choroidal hemangioma.
PMCID: PMC4033500  PMID: 24955093
21.  Valsalva Retinopathy Associated with Sexual Activity 
Case Reports in Medicine  2014;2014:524286.
A 54-year-old healthy male presented complaining of sudden loss of vision in the right eye. Initial visual acuity was counting fingers. The patient's acute vision loss developed after sexual activity. Color fundus photos and fluorescein angiography were performed showing a large subinternal limiting membrane hemorrhage in the macular area. A 23-gauge sutureless pars plana vitrectomy with brilliant blue assisted internal limiting membrane peeling was performed with best-corrected visual acuity recovery to 20/50 at 6 months of followup.
PMCID: PMC4026870  PMID: 24876844
22.  Bortezomib Induced Hepatitis B Reactivation 
Case Reports in Medicine  2014;2014:964082.
Background. It has recently been reported that hepatitis B (HBV) reactivation often occurs after the use of rituximab and stem cell transplantation in patients with lymphoma who are hepatitis B surface antigen (HBsAg) negative. However, clinical data on HBV reactivation in multiple myeloma (MM) is limited to only a few reported cases. Bortezomib and lenalidomide have remarkable activity in MM with manageable toxicity profiles, but reactivation of viral infections may emerge as a problem. We present a case of MM that developed HBV reactivation after bortezomib and lenalidomide therapy. Case Report. A 73-year-old female with a history of marginal cell lymphoma was monitored without requiring therapy. In 2009, she developed MM, presenting as a plasmacytoma requiring vertebral decompression and focal radiation. While receiving radiation she developed renal failure and was started on bortezomib and liposomal doxorubicin. After a transient response to 5 cycles, treatment was switched to lenalidomide. Preceding therapy initiation, her serology indicated resolved infection. Serial monitoring for HBV displayed seroconversion one month after change in therapy. Conclusion. Bortezomib associated late HBV reactivation appears to be a unique event that requires further confirmation and brings to discussion whether hepatitis B core positive individuals would benefit from monitoring of HBV activation while on therapy.
PMCID: PMC4021848  PMID: 24876846
23.  Extreme REM Rebound during Continuous Positive Airway Pressure Titration for Obstructive Sleep Apnea in a Depressed Patient 
Case Reports in Medicine  2014;2014:292181.
A 20% increase in REM sleep duration has been proposed as a threshold to identify REM rebound in patients with obstructive sleep apnea (OSA) who start continuous positive airway pressure (CPAP) treatment. We describe the case of one patient with OSA who showed an unexpectedly high degree of REM rebound during titration of CPAP. A 34-year-old man was diagnosed with OSA. He remained untreated for many years, during which he developed systemic hypertension, depression, and severe daytime somnolence. When he was reevaluated sixteen years later, his Epworth sleepiness score was 18, and his OSA had greatly worsened (apnea/hypopnea index: 47, lowest nocturnal saturation: 57%). He underwent a successful CPAP titration during nocturnal polysomnography. Electroencephalographic analysis of the sleep recording revealed a huge amount of REM sleep, accounting for 72% of the total sleep time. When asked, the patient referred that he had suddenly interrupted paroxetine assumption three days before the polysomnography. The very large REM rebound observed in this patient could be due to additional effects of initiation of CPAP therapy and suspension of antidepressive treatment. This case does not report any dangerous consequence, but sudden antidepressive withdrawal could be dangerous for patients with OSA who develop hypoventilation during REM sleep with CPAP application.
PMCID: PMC4024427  PMID: 24876843
24.  Pars Plana Vitrectomy Combined with Focal Endolaser Photocoagulation for Idiopathic Macular Telangiectasia 
Case Reports in Medicine  2014;2014:786578.
Background. To report the outcome of pars plana vitrectomy (PPV) combined with intraoperative endolaser focal photocoagulation (PC) on eyes with idiopathic macular telangiectasis (MacTel) type 1. Methods. This was a retrospective study of two female patients with MacTel type 1 who were resistant to focal photocoagulation, sub-Tenon triamcinolone injection, and/or antiangiogenic drugs. The best-corrected visual acuity (BCVA) was determined, and fluorescein angiography (FA) and spectral domain optical coherence tomography (SD-OCT) were performed before and after surgery for up to 19 months. Results. After surgery, the BCVA gradually improved from 20/100 to 20/20 at 19 months in Case 1 and from 20/50 to 20/13 at 13 months in Case 2. Fluorescein angiography (FA) showed leakage at the late phase, and OCT showed that the cystoid macular edema was resolved and the fovea was considerably thinner postoperatively. Conclusion. Patients with MacTel type 1 who are refractory to the other types of treatments can benefit from PPV combined with intraoperative endolaser focal PC with functional and morphological improvements.
PMCID: PMC4021993  PMID: 24876845
25.  Purulent Pericarditis after Liver Abscess: A Case Report 
Case Reports in Medicine  2014;2014:735478.
We present the case of a 49-year-old woman, with previous clinical antecedents of recent hepatic metastasis, who was admitted to the ICU due to respiratory failure and hemodynamic instability. She was found to have purulent pericarditis complicated by pericardial tamponade and pleural effusion, as well as surgical site infection, which was the origin of the disease. Cultures of the surgical wound and the pericardial effusion were positive for Enterococcus faecalis and Escherichia coli. A pericardial tap was performed and the intra-abdominal abscess was surgically drained. Pleural effusion was also evacuated. She received antibiotic treatment and recovered successfully. The only after-effect was a well-tolerated effusive-constrictive pericarditis.
PMCID: PMC4020547  PMID: 24872819

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