A 55-year-old man, lawyer, was admitted to the hospital because of abdominal pain located in the epigastrium and right hypochondrium, intermittent night sweats, and 3 kg involuntary weight loss over the preceding 8 months. Approximately 5 years earlier severe aortic valve stenosis, with left ventricular hypertrophy, paroxysmal atrial fibrillation, and heart failure (class III NYHA) were diagnosed, and aortic valve replacement was performed using a biological prosthetic valve. Furthermore, the patient was an alcohol user (70 g/day) and had endoscopically diagnosed chronic antral gastritis. He lived in an urban area but, approximately 18 months before admission, he traveled to Nepal living in settings with poor hygienic standards. Daily medications included omeprazole 20 mg.
Approximately one year before admission, the patient began to have intermittent, profuse night sweats associated with abdominal pain located in the epigastrium, and right hypochondrium. He reported no fever or chills. No remarks on routine physical examination performed by his attending physician were made, and summarizes the results of prescribed laboratory tests. Serologies for human immunodeficiency virus (HIV), hepatitis B virus (HBV), hepatitis C virus (HCV), and VDRL were negative. Serologies for cytomegalovirus (CMV) and Epstein Barr virus (EBV) showed past infections. Two blood cultures were negative. A transthoracic echocardiography detected hemodynamically well-functioning aortic valve prosthesis, ascendant aortic dilatation, and no valve vegetations. An abdominal ultrasound revealed a biliary cyst of the liver. His physician eventually decided to prescribe sertraline for depression.
Four months before admission, the patient was still complaining of abdominal pain, with the same characteristics and was referred to our out-patient clinic. Laboratory tests were repeated (), protein electrophoresis was performed, and results were within normal range.
Meanwhile, during a work trip to Brazil, the patient developed lower limbs leukocytoclastic vasculitis ().
Due to worsening of intermittent night sweats, persistence of abdominal pain, and 3 kg involuntary weight loss over the preceding 8 months with no fever, the patient was admitted to the hospital.
On examination, the patient was in good condition, the temperature was 37.1°C, the pulse rate 75 beats per minute, the blood pressure was 130/75 mmHg, and respiratory rate was 15 per minute. The oxygen saturation in ambient air was 98%, good oral and dental health. There was no superficial lymphadenopathy. There was no jugular venous distension. Auscultation of the heart revealed a mechanical second heart sound, a grade II/VI systolic murmur in the aortic area. No chest rales were detected. There were no conjunctival hemorrhages, Osler's nodes, Janeway's lesions, Roth's spots, splinter hemorrhages, or peripheral edema.
The electrocardiogram showed a normal sinus rhythm with nonspecific repolarization abnormalities.
Routine laboratory tests () were performed and revealed hemoglobin 12.8 g/L [13.5–17.5], platelet count 139,000 per mm3 [150,000–450,000], aspartate aminotransferase (AST) 61 U/L [<50 U/L], alanine aminotransferase (ALT) 73 [<50 U/L], alkaline phosphatase (ALP) 110 [<120 U/L], ferritin 418 ng/mL [24–336], C-reactive protein (CRP) 4.1 [<5.0 mg/L], erythrocyte sedimentation rate (ESR) 15 mm/hr [<20], and rheumatoid factor (RF) 318 U/mL [<14]. Electrolyte panel and urinalyses were normal. Serologies for HIV, HBV, HCV, Schistosoma, Borrelia, Rickettsia, Leishmania, and VDRL were negative. Serologies for CMV, EBV, and Toxoplasmose showed past infections. Widal and Huddlesson reactions and research of Plasmodium sppwere negative as well. Two sets of blood culture specimens exhibited no growth. Protein electrophoresis showed IgG-K/λ monoclonal hypergammaglobulinemia. Abdominal ultrasound detected mild hepatosplenomegaly. Thoracic-abdominal-pelvic computer tomography disclosed gastric wall calcification and confirmed hepatosplenomegaly. To rule out endocarditis, transesophageal echocardiography was performed. Two very small echodense images suggestive of surgery-related alterations versus fibrosis were detected on the aortic cups (Figures and ). Fundus examination showed no retinal abnormalities.
Liver biopsy revealed infectious hepatitis without pathognomonic features () and with no fibrin ring granulomas described in Q fever.
EDTA-whole blood and paraffin-included liver biopsy were then sent to CEVDI/INSA for C. burnetii laboratory diagnosis. The serologic evaluation was performed by Indirect Immunofluorescence Assay (IFA) using the commercial C. burnetii I+II IgG/A/M Immunofluorescence kit (Vircell Microbiologists, Granada, Spain). It revealed a high anti-phase I IgG with an end-point titer of 204,800 suggestive of chronic Q fever (anti-phase I IgG ≥800 was the reference cut-off, for results). C. burnetii active infection was also confirmed by molecular testing. For this purpose, genomic DNA was extracted from buffy coat and liver tissue using DNeasy blood & tissue kit (Qiagen GmbH, Hilden, Germany). The presence of C. burnetii DNA was screened by polymerase chain reaction (PCR) in a nested reaction using the primers pairs Trans1-Trans2 and Trans3-Trans4 that target a transposon-like repetitive region in the agents genome []. Specific DNA was detected in liver tissue but not in blood, and C. burnetii identity was confirmed by amplicon sequencing. The quality of DNA in both samples was previously verified by amplification of human β-actin gene as internal control. Moreover, the absence of bacteria in blood sample was reinforced by a negative result also obtained for the isolation attempt. In fact, an additional buffy coat aliquot was inoculated in DH82 cells, routinely used in CEVDI for C. burnetii isolation, and after a standard 60-day incubation at 37°C and 5% CO2 atmosphere in BSL3 condition, no bacteria growth could be detected. The paraffin processing of the liver sample limited its utility for agents isolation.
Final diagnosis of chronic Q fever was made, with histologically documented infectious hepatitis and endocarditis by modified Duke's criteria []. Our patient met one of the major criterion of Q fever endocarditis (C. burnetii serology with anti-phase I IgG titer >800) and three minor criteria (predisposing heart condition with biological prosthetic aortic valve, immunological phenomena with RF positive, vascular phenomena with leukocytoclastic vasculitis). Furthermore, we probably may consider the valvular alterations detected by transesophageal echocardiography as a minor criterion (echocardiographic findings not meeting a major criterion).
Antimicrobial therapy with doxycycline 100 mg bid, and hydroxychloroquine 200 mg tid was started and the patient became asymptomatic within 8 days.
The patient is still being followed in our out-patient clinic and he is adhering to treatment. He remains well and his weight gradually returned to normal (75 kg-Bode Mass Index 23,1), 8 months after the diagnosis. The titers of antibodies to C. burnetii have been falling in response to therapy. Results of the periodical serological tests are summarized in , together with results of the laboratory tests ().


A 26-year-old male patient was diagnosed as AML-M2 in 1995. Following an induction regimen he achieved complete remission (CR) and received an allogeneic peripheral blood stem cell transplantation from his 37-year-old HLA identical brother in 1997. The conditioning regimen consisted of busulfan and cyclophosphamide, and the GVHD prophylaxis was done with cyclosporin and short-term methotrexate. He did not develop any acute or chronic GVHD and remained in CR with complete donor-type chimerism until 2008 when he was admitted to the neurosurgery clinic with back pain. The vertebral MRI revealed a tumor invading the 6th thoracic vertebrae causing pathological fracture. The tumor was completely excised, and the pathological evaluation was consistent with CD38++ and CD117+ atypical plasma cell infiltration with kappa monoclonality. He had IgG kappa monoclonal gammopathy on immune electrophoresis and an elevated erythrocyte sedimentation rate along with a mild anemia and normal renal function tests. Bone marrow (BM) examination revealed 9% plasma cells with CD38+CD138+CD19−CD56−CD44+CD28−CD20− immunophenotype and kappa predominance. The cytogenetic study did not detect any abnormality. Bone marrow chimerism analysis was consistent with 100% donor type in both T-and non-T-cell lineages. Chimerism study was also done with the DNA extracted from the plasmacytoma and revealed 18% donor and 82% recipient cells (). The presence of EBV could not be demonstrated in BM or plasmacytoma by in-situ hybridization for EBV early RNA as well as PCR analysis of EBV DNA. The donor was also found to be negative for signs of secretory paraproteinemia. The patient received 3000 cGy local radiotherapy to T5–T7 level of the spine and high-dose oral dexamethasone. After attaining a CR, it was decided to perform an allogeneic HCT without immunosuppression, instead of the general approach of autologous transplantation, since he had complete donor type of chimerism. Following a nonmyeloablative conditioning with fludarabine and melphalan a hematologic engraftment was achieved. The BM and blood examinations revealed CR for myeloma and a continued complete donor-type chimerism. The patient developed a grade 2 acute GVHD of skin which was controlled with steroids, but acute grade 3 gastrointestinal GVHD required the addition of cyclosporin. Later immunosuppression could be tapered off completely without causing rebound GVHD. He continued in CR until one year after transplantation when he developed progressive mucor sinusitis and infectious meningitis and eventually died.


A 65-year-old woman presented with a mass 6 cm in diameter located on her right frontoparietal region. The mass had appeared approximately three months earlier and had grown rapidly. The patient's history included resected colorectal adenocarcinoma followed by general surgery and was disease-free for five years. She was referred to us because the swelling and pain indicated a sebaceous cyst or a benign soft tissue tumor (). The mass was indurated, round, and adherent to the deep planes. Mild alopecia and skin expansion due to the mass were observed on her anterior scalp. The patient complained of tenderness and mild pain when the mass was palpated. Our clinical differential diagnoses included sebaceous cyst, lipoma, or localized soft tissue tumors. But the plain radiographs and the computerized tomography demonstrated a destructive mass which was eroding the bone over the right frontoparietal region ().
A transverse incision and a meticulous blunt dissection that were performed under local anesthesia exposed a rubbery soft tissue mass extending to the outer table of the skull. The mass was dissected with care. However, we noticed calvarial bone destruction with an exposed duramater and lytic lesions 2 × 3 cm in size around the calvarial defect. The mass was completely resected. The mass was measured to be 5.5 × 5.0 × 2.0 cm (). The damaged bone sites were resected with a 1 cm security margin. A section of the duramater measuring 2 × 3 cm was removed and the defect was reconstructed using a tensor fascia graft by the brain surgeons. Hemostasis was achieved using electrocautery and bone wax. A bolster dressing was applied following the primary closure of the skin incision. The specimen was sent for histopathological analysis. The patient was monitored closely for one day and was discharged the next day.
Histopathologic findings revealed many low differentiated adenoid, and apoptotic and necrotic spaces lined with atypical epithelial islands with vesicular nuclei. At high-power magnification, the specimens showed cytologic atypia with pleomorphic and hyperchromatic nuclei. Immunohistochemical analysis was positive for carcinoembryonic antigen (CEA), villin, and cytokeratins 7 and 20. The specimens were negative for vimentin, chromogranin, thyroglobulin, thyroid transcription factor 1 (TTF-1), and CDX2. The final diagnosis was metastatic low-differentiated adenocarcinoma compatible with the immunophenotype of colorectal adenocarcinoma (Figures , , and ). After the histopathological evaluation revealed metastatic adenocarcinoma, the patient was referred to the medical oncology department.


A 58-year-old female had lumbar pain and shortness of breath at the time of admission. Medical history revealed the patient being under medical treatment for chronic obstructive lung disease. A slight limitation in lumbar movements was determined by physical examination. Routine laboratory tests were normal. Further radiological evaluation included ultrasonography (US), color Doppler ultrasonography (CDUS), thoracoabdominal magnetic resonance imaging (MRI), and magnetic resonance cholangiopancreatography (MRCP). The balanced turbo field echo (BTFE-BH SENSE) sequence on MRI (Figures and ) revealed following findings: the aorta, aortic arc, and apex of the heart were located on the right-hand side, and the inferior vena cava, stomach, and spleen were located on the left-hand side according to the midline. The liver was located in the midline, but it was left-dominant liver. The position of the gallbladder was right-hand sided under the liver. The pancreas was located on its normal position in the midline. The spleen had three foci (polysplenia) and was in a settled position on the left-hand side under the liver. The right renal vein which was retroaortic has passed posterior to the aorta and drained into the inferior vena cava. The small and large intestines seemed to have some degree of malrotation. It was observed that the large and small intestines were predominantly settled in lower right region and upper left region of the abdomen, respectively. Color Doppler ultrasonography showed that the inferior vena cava and aorta flows were on the opposite side of the normal location (). On MRCP, the liver was observed to be left dominant located, and common bile duct was lying towards the inferomedial from the left-hand side.


In 2005, a 42-year-old Brazilian woman presented to an eye doctor with a visual impairment, and pappiledema. Upon neurological examination, she was diagnosed with communicating hydrocephalus. A ventriculo-peritoneal shunt was implanted and the hydrocephalus improved. Neuroendoscopic surgery showed multiple cysts in the subarachnoidal space, and histopathological examination indicated cestode-derived tissue. Serology for cysticercosis was strongly positive, but copro-multiplex PCR was negative for taeniasis. The patient was treated with praziquantel at first and then with albendazole. Post-treatment serology remained positive.


A 38-year-old Japanese man expelled a single tapeworm in 2000. He was treated with pyrantel pamoate, a drug used primarily for nematode parasites. In 2004, he complained of headaches and multiple cystic lesions were found on brain computed tomography (CT). However, at this time, no medical or surgical treatment was initiated because all of the cysts were deemed dead based on the CT image and the patient's normal eosinophil value. In 2009, he complained of a narrowing field of vision. Serology showed strong positive responses, but copro-LAMP was negative. The patient was treated with albendazole in 2010, and follow-up is ongoing.


A 31-year-old Japanese man noticed a subcutaneous nodule on his neck in December 2009. When the number of nodules increased by May 2010, brain CT and magnetic resonance imaging (MRI) showed multiple cystic lesions in the brain and serology showed strong positive responses. Tapeworm proglottids were expelled in his stools at the beginning of July 2010 and were identified as T. solium by multiplex PCR. He remembered he had found a similar white noodle-like substance in 2009, but he did not care about it. Taeniasis was treated using Gastrografin (diatrizoate meglumine and diatrizoate sodium) at Mie University hospital in Mie, Japan on 14 July, but no worms were expelled. However, T. solium infection was confirmed by copro-LAMP and Taenia eggs were identified microscopically. On July 28th 2010, the patient expelled additional worms. Then he was treated with Gastrografin and albendazole in August 2010, but tapeworm(s) were not expelled. However, it was considered that the patient became free from T. solium tapeworm after he found the worm by himself on 28th July, as copro-LAMP was negative until December 2010. In contrast, the subcutaneous and intracranial nodules disappeared within 4 weeks after the treatment.
The patient's travel history indicated that he had worked for one month in a rural area 150 km from Delhi, India on 3 separate occasions. The first, second and third visits were March-April 2008, October-November 2008 and March-April 2010, respectively. Based on this information, it was suspected that he acquired the parasite during his first or second stay in India. A mtDNA gene (chromosome c oxidase subunit 1) of the obtained proglottids was sequenced according to previous research [], and was determined to be identical to the Indian haplotype of T. solium (GenBank Accession Number: ). Due to the risk of the patient contaminating his living environment with parasite eggs while infected with the intestinal form of T. solium, his wife and 4 year old son were tested serologically for cysticercosis. In addition, 41 colleagues who had also travelled to India since 2007 were tested serologically for cysticercosis, with 39 of these individuals also tested for taeniasis using copro-LAMP. To date, none of these individuals have tested positive for either cysticercosis or taeniasis. This case is notable as a rare imported case of T. solium taeniasis/cysticercosis in Japan.


A Caucasian 18-month-old girl, born to an Italian family and raised in Italy, with a high-risk acute lymphoblastic leukemia (ALL) was being treated according to the AIEOP-BFM ALL 2000 protocol. After the third dose of VCR (1.5 mg/m2) she developed isolated hoarseness, but stridor appeared soon after the fourth dose of VCR (last dose of induction phase). A flexible fiber-optic endoscope study showed a bilateral VCP (Figure ). Electromyography revealed a predominantly axonal motor neuropathy involving above the lower extremities. A nerve conduction study of the larynx was not performed because the association of VCR administration with significant respiratory symptoms and immobility of the vocal folds, clearly shown by the fiber-optic endoscope, allowed us to make a definite diagnosis. No side effects of VCR other than VCP were found. Her stridor started to improve within the first seven days and both stridor and hoarseness completely resolved 28 days after the onset of palsy: a repeat laryngoscopy study showed normal vocal cord mobility.
Following two consolidation blocks it was decided to administer VCR at full dose (1.5 mg/m2) first, and completely omit the second dose (scheduled just five days later): no side effect was noted. The first reinduction protocol II started 22 days after completion of consolidation block therapy and the four weekly doses of VCR were uneventfully administered with a one-third reduction (1 mg/m2).
After four weeks of interim maintenance the second reinduction protocol II was scheduled and the first weekly dose of VCR was again administered at 1 mg/m2 without any problems; then our patient received glutamic acid 1.5 g daily orally in three divided doses and, one week later, a bolus injection of VCR was administered at full dose. Three days later she developed an intermittent hoarse voice lasting about 72 hours. Moreover, she manifested reduction of deep tendon reflexes, neuritic calf pain (treated with oral acetaminophen) and foot drop; the pain and foot drop resolved after about 24 hours. The following weekly VCR was omitted and glutamic acid was stopped after 10 days of overall treatment. The fourth and last dose of VCR was administered again at 1 mg/m2 and was well tolerated. She is now in complete remission and on maintenance therapy.


A 26-year-old Caucasian woman who had mild lung disease presented to our facility. Her pre-partum forced expiratory volume in one second (FEV) was 2.62 L (87% predicted). Her pre-partum body mass index (BMI) was 24. She was chronically infected with Pseudomonas aeruginosa. In addition, she was pancreatically insufficient and had CF-related diabetes treated with insulin. Her glycemic control was acceptable with a hemoglobin A1C (HbA1C) level of 6.1%. She did not have microalbuminuria as estimated by a spot albumin:creatinine ratio. Eight years previously she had delivered a healthy child via a normal vaginal delivery after spontaneous rupture of membranes at 36 weeks’ gestation. The pregnancy had been uncomplicated.
Her second pregnancy was unplanned and she had conceived with a new partner. Following confirmation of the pregnancy, she was referred to a high-risk pregnancy clinic where she was reviewed regularly by a multidisciplinary team supervised by an obstetrician, a respiratory physician and an endocrinologist. The first two trimesters proceeded without significant medical complication. During the 26th week of pregnancy, a routine out-patient review revealed a fall in her lung function (FEV1 2.13 L, approximately 71% predicted) accompanied by increased respiratory symptoms. Her blood pressure was noted to be slightly elevated at 118/87. A hospital admission for treatment of her fall in lung function was declined and she was given a two-week course of oral ciprofloxacin at a dose of 750 mg twice a day. This was tolerated poorly due to nausea.
Two weeks later, in the 28th week of pregnancy, she developed worsening nausea and further exacerbation of her respiratory symptoms with some accompanying left upper quadrant discomfort. Her blood pressure was 123/90. The results of a urinary test strip screening for proteinuria were negative. A hospital admission was again declined and treatment with oral ciprofloxacin for a presumed mild pulmonary exacerbation was continued, and she was prescribed oral metoclopramide, with outpatient follow up arranged in one week. She was advised to present to hospital sooner in the event of clinical deterioration.
Our patient re-presented one week later with increasing nausea and a persistent impairment of lung function (FEV1 2.09; 69% predicted). She was admitted to hospital. At this time, her blood pressure was 153/89. Laboratory investigation results revealed hemoglobin of 12.3 g/dL, platelet count of 101 × 109 cells/ml (normal range 150 to 400 × 109 cells/ml), serum alanine transaminase (ALT) of 179 mmol/L (normal range <65 mmol/L), lactate dehydrogenase (LDH) of 279 mmol/L (normal range <240 mmol/L) and urate of 523 mmol/L (normal range 150 to 400 mmol/L). There was no significant proteinuria as assessed by a urinary protein/creatinine ratio and this remained unchanged throughout the admission. Similarly, serial assessment of her coagulation and renal function failed to reveal any abnormality. On admission, a fetal ultrasound was within normal limits.
A diagnosis of pre-eclampsia with some features of HELLP syndrome was made, in the setting of a respiratory exacerbation. In addition to her regular medications, she was put on intravenous aztreonam at a dose of 2 g every six hours. She was given two 11.4 mg doses of intramuscular betamethasone at 12-hour intervals to assist with fetal lung maturation. Aminoglycosides were not given to avoid any potential harm to the fetal kidneys. Over the next five days she remained hypertensive with a systolic blood pressure ranging from 150 to 190 mmHg, but her platelet count increased to 239 × 109 cells/ml and the results of liver function tests (LFTs) progressively normalized. However, on the fifth day there was a precipitous drop in platelet count to 62 × 109 cells/ml and an increase in ALT to 118 mmol/L, accompanied by a fall in hemoglobin to 8.8 g/dL with red cell fragments observed on blood films taken at this time. A diagnosis of HELLP syndrome was made and a semi-elective caesarean section was performed 10 hours later under spinal anesthesia. HELLP-specific treatment, in particular magnesium sulfate was not administered. A healthy baby girl was delivered. Her APGAR scores were 9 and 9 at one and five minutes, respectively. All of our patient’s blood parameters normalized the day following birth of her child. Our patient recovered without further medical complication. Four years later, our patient’s lung function remains stable, although she continues to have intermittent exacerbations of her bronchiectasis. Our patient’s child has reached normal age-specific milestones.


A two days old male baby weighing 2.5 kg presented with abdominal distension, failure to pass meconium and biliary emesis since birth. The baby was born by spontaneous vaginal delivery at home. According to the mother the patient was born with a distended abdomen and passed white pellets per rectally. The abdominal distension gradually worsened with bilious vomiting after every attempt at feeding. General physical examination revealed a lethargic and ill looking baby with obvious respiratory distress and abdominal distension. He was febrile with temperature of 100 °F; respiratory rate 45/min, and pulse 150/min. Bowel loops were visible. A per-rectal examination revealed meconium beads. A preoperative diagnosis of neonatal intestinal obstruction secondary to meconium ileus, with a differential of distal intestinal atresia, was made.
The newborn was resuscitated with intravenous infusion. A nasogastric tube was passed and gastric aspiration done. The neonate was given injection Vit. K and intravenous antibiotics started. X-ray abdomen showed dilated bowel loops. Contrast x-rays with gastrografin enema delineated a small caliber colon with filling defects as of meconium beads (Fig. ). As condition of the baby did not improve an exploratory laparotomy was performed. At laparotomy volvulus of distended small bowel found. The colour of the involved segment was dark and appeared congested (Fig. ). The volvulus was corrected by untwisting the mesentery. The involved gut was hugely distended due to the presence of thick tenacious meconium. The thick meconium was concentrated in distal small bowel whereas the colon was packed with small beads. An enterotomy was made at the most distended portion (distal ileum) and irrigated with diluted gastrografin. Gentle milking was then performed to remove the meconium. The large gut also washed in the similar way. Bishop Koop chimney was made after resecting a small portion of small bowel which was of doubtful viability.
The postoperative recovery was uneventful. NG tube was removed on 4th post-op day with oral feed on following day and discharged on 7th postoperative day. The patient has an uneventful follow up after Bishop-Koop stoma closure (at the age of 6 months).


A 61-year-old male with history of Alzheimer's dementia, diagnosed at the age of 50, was admitted for worsening mental status and paranoid behavior. He was started on olanzapine a few weeks prior to this admission. He was found to be lethargic, drowsy, lying down in the bed motionless. His home medications included donepezil, alprazolam, and trazodone in addition to recently started olanzapine. On physical examination he had a fever of 102.1°F, heart rate—122 beats/minute, blood pressure—106/77 mm of Hg, and respiratory rate—22 breaths/minute with 100% oxygen saturation on room air. He was drowsy lying down in bed, only opening eye to repeated verbal commands. He had opisthotonus, with diffuse rigidity; reflexes were not well elicitable in view of diffuse rigidity. The rest of system examination was noncontributory.
Laboratory data showed hemoglobin level of 12.5 mg/dL (reference range—13.5–17.5 g/dL), WBC—12.5 × 103/μL (reference range—4.5–10.8 × 103/μL), platelet—253 × 109/L (reference range—150–450 K/μL), and creatinine kinase (CK)—6400 units/L (reference range—38–174 units/L). Blood cultures were negative; CT scan of head, abdomen, and thorax was normal; Cerebrospinal fluid analysis was normal and the urine drug screen was negative. In view of rigidity, fever and increased CK along with the history of recently added olanzapine, a diagnosis of neuroleptic malignant syndrome (NMS) was made. He was managed in the intensive care unit and in addition to hydration was treated with intravenous dantrolene (which was later changed to oral bromocriptine). Throughout his stay in the hospital he was not physically restrained and was kept on deep venous thrombosis (DVT) prophylaxis with enoxaparin. On the fourth day of admission, the patient developed swelling of arms, neck, and right lower limb. Doppler studies revealed venous thrombosis involving the right axillary, right subclavian, right internal jugular vein, left axillary vein, and right common femoral vein. The patient was thus treated with intravenous heparin (with warfarin bridging). The standard workup for new onset DVT was negative. He had normal levels of Antithrombin III, Protein C, and Protein S. In addition he was negative for Factor V Leiden, ANA, and Lupus Anticoagulant.
Considering the temporal sequence of events and no previous history of DVT, either NMS or olanzapine or a combination of both could be implicated as the possible etiology for the extensive venous thrombosis. At time of discharge he was back to his baseline functional state. He was discharged on warfarin for a period of six months.


We describe the case of a 38-year-old woman with diagnosis of CML Phi(+) in chronic phase whose main aim was to have a baby and plannified her pregnancy stopping imatinib to be out risk. She achieved a safe pregnancy and delivery maintaining an optimal molecular response through it.
In June 2000, when she was 27 years old, leucocytosis with left deviation was confirmed, during a routine check-up exam. Her physical examination was normal. Peripheral blood (PB) count findings were as follows: white blood counts (WBC) 127 × 109/L with 1% blasts, 18% myelocytes, 12% metamyelocytes, 12% bands, 51% neutrophils, 4% lymphocytes, 2% monocytes, hemoglobin 12.9 g/dL, and platelet count 131 × 109/L. Renal and liver function tests were normal. Cytogenetic and molecular tests confirmed 100% Phi(+) in all analyzed metaphases and presence of BCR-ABL 4 rearrangement by polymerase chain reaction (PCR). Search for HLA compatible donor with her sister was unsuccessful. She had a low-risk Sokal score and her performance status was 0 at diagnosis. She was first treated at another institution and given IFN alpha 5.000.000 U/day associated with cytarabine 30 mg/day for 10 days every 28 days. She obtained complete hematologic and cytogenetic response at 2 and 12 months, respectively, and was maintained over time. In July 2005, she was referred to FUNDALEU, and started imatinib 400 mg/day with excellent tolerance and adherence to medication. She had been monitored by fluorescence in situ hybridisation analysis (FISH) every 6 months with persistence of CCyR. She was advised about imatinib teratogenicity deciding to use contraceptive methods.
Since 2007, real-time quantitative PCR (RQ-PCR) started to be performed under the standardization program. In November 2007, she was first tested by RQ-PCR showing achievement of MMR and monitored every 6 months showing a molecular kinetic stability through time with persistent MMR/CMR up to present time. In kinetics of molecular response is described. In September 2009, she expressed her wish to have a baby asking if a future pregnancy could be possible. She received a long explanation of what the literature describes about possible teratogenic effects imatinib causes to the fetus while mother is under treatment and also the possible but unknown risks for progression if interrupting treatment at this stage of the disease. After that she planned the possibility of being carried out. She was then 36 years old, and due to that she was referred to a consultation with a fertility specialist. The first step was to study both, the patient and her husband, in order to detect any abnormality that could potentially delay the achievement of pregnancy before the interruption of imatinib. Basic fertility workup: spermogram, gynaecologic physical examination, hysterosalpingography, basal FSH, LH, estradiol, prolactin and 5 thyroid hormone levels, and cervical and vaginal cultures, as well as transvaginal ultrasound were, between normal limits.
After being told of the risks described above, she signed the informed consent and imatinib was withhold on January 20th, 2010. Planification of monthly visits with blood counts were required associated to RQ-PCR every 3 months. As she had not become pregnant the 2nd month after the interruption of imatinib and with consensus of the hematologist and fertility specialist, it was decided to start ovarian stimulation for intrauterine insemination; although the couple was considered presumably fertile. Ovarian stimulation was performed with 100 mg daily of clomiphene citrate from day 5 to 9 of the menstrual cycle, followed by daily subcutaneous application of 75 I.U. of HMG. When three follicles of 20, 18, and 17 mm. in mean diameter where observed by transvaginal ultrasound, ovulation was triggered with 10000 I.U. of hCG. Thirty-six hours later an intrauterine insemination with 53 × 106 highly motile spermatozoa after swim-up preparation of the semen sample was performed. As a result of the procedure she achieved a singleton pregnancy. She required hospitalization between weeks 23 and 24 for suspected preterm labour, receiving corticosteroids after completion of week 24, to prevent fetal lung maturation. Although treatment was interrupted, she persisted on CHR and MMR during all the pregnancy. Labour started spontaneously in December 2010, at week 39, requiring an emergency caesarean section because of fetal distress. A healthy girl was born, with Apgar Score of 9/10, weight of 3.010 grs, and measuring 49 cm in length and 33 cm of cephalic diameter. Negative DAT and physiologic jaundice. Patient's molecular evaluation after delivery showed MMR persistency, so breastfeeding was permitted. Having breastfed for 3 months, she reinitiated imatinib 400 mg day on April 2011. The girl's growth and development had been normal to date.
Data about imatinib and breastfeeding in humans is still limited and the effects of chronic exposure of infants to imatinib are not known. Its metabolites are excreted in the milk of female rats when given at 100 mg/kg daily with higher concentrations than in plasma. Substantial accumulations of imatinib into breast milk were observed when measured in women taking imatinib postpartum, and due to possible adverse reactions, breastfeeding should be not be permitted [].


A 46-year-old morbidly obese Caucasian female was referred by her general practitioner (GP) to rheumatologist for further evaluation of 25 kilograms weight loss over a period of one year associated with persistently elevated ESR of 60 mm/hr. Her history was remarkable for hypertension, depression, and GORD. Medications disclosed at initial consultation included verapamil SR 240 mg/day for a well-controlled hypertension. Clinical examination at presentation revealed an obese woman with a body mass index of 58 kg/m2 without clinical features of Cushing's syndrome. The rest of the physical examination was normal. All rheumatologic and vasculitic studies were unremarkable. As part of the work-up, an ultrasound and CT scan showed pancreatic head mass measured 3.6 × 2.4 cm diameter. The radiological differentials were cystoadenoma and adenocarcinoma. Followup liver function test showed no evidence of biliary obstruction. Joint gastroenterological and endocrine biochemical assessment revealed normal serum glucose, glucagons, and vasoactive intestinal peptide. Other tumor markers were undetectable. Further investigations included normal findings for cortisol rhythm and low dose dexamethasone suppression. Urinary catecholamine and 5-hydroxy indole acetic acid excretion did not suggest elevated hormonal activity. An initial serum CgA measured by enzyme-linked immunosorbent assay (0.5 U/L detection limit; Dako, Denmark) was noted to be moderately elevated 46.0 U/L (normal < 17.2 U/L) and rose to 176 U/L in 4 months (Figures and ). Simultaneous serum gastrin level was elevated twice upper limit of normal 198 ng/L (normal < 100 ng/L). Further clinical review highlighted previously undisclosed usage of PPI, rabeprazole 40 mg once daily dating back to the twelve-month period of work-up. This medication was then subsequently suspended resulting in normalization of CgA () and subsequent 6-month followup confirmed consistently undetectable serum CgA levels while off the PPI. 131I-MIBG scintigraphy did not show pathologic isotope accumulation and serial CT scan of abdomen revealed no increase in size of the pancreatic mass. Endoscopic ultrasound-guided fine needle biopsy confirmed the lesion to be a benign pancreatic hemorrhagic cyst.


A 36-year-old Japanese man, with a history of presumed Crohn’s disease at age 16 years, on the basis of peri-anal fistula with compatible histopathological features and spontaneous healing without medical intervention, presented to our facility with a 10-day history of fever (39°C to 40°C), malaise and generalized lymphadenopathy. He also had a history of photosensitivity and erythema on the cheeks and nose over the past five years, which had worsened over the previous two days. Our patient had no history of smoking or asbestos exposure. On examination, he was febrile and had tachypnea. He had a bilateral malar rash, tender cervical lymphadenopathy, swelling of parotid and submandibular glands, and arthritis affecting the bilateral metacarpophalangeal joints of the hands. He also had a dry mouth and dry eyes, which were confirmed by a positive Schirmer’s test (<5mm per five minutes, bilaterally). Auscultation of the heart and lungs was normal. His abdomen was remarkable for moderate hepatomegaly and splenomegaly.
On admission, his laboratory test results showed: leukocytosis (white blood cells 13,090 cells/μL; lymphocytes 57.4%, atypical lymphocytes 10.0%); elevated liver enzymes (alanine aminotransferase 202U/L (normal range 10 to 40U/L), aspartate aminotransferase 172U/L (normal range 13 to 30U/L), alkaline phosphatase 1332IU/L (normal range 100 to 320IU/L), γ glutamyltranspeptidase 223IU/L (normal range 5 to 60IU/L), lactate dehydrogenase 561IU/L (normal range 110 to 210IU/L)); and normal renal function and urine analysis with serum angiotensin-converting enzyme 16.4U/L (normal range 7 to 25U/L). Serum serological tests were positive for anti-nuclear antibody 1:80 (normal value <1:40), double-stranded DNA (dsDNA) antibodies 22IU/mL (normal value <10IU/mL), and anti-Sjögren syndrome antigen A (SS-A)/Ro antibody >500 (normal value <10).
Virological test results for human immunodeficiency virus antibody, cytomegalovirus antigen and antibody, hepatitis B surface antigen and antibody, hepatitis C virus antibody were negative, but our patient was positive for IgG and IgM to EBV viral capsid antigen and IgG to EBV early antigen. Our patient was negative for IgG to Epstein-Barr nuclear antigen (EBNA). A chest X-ray was clear and computed tomography (CT) revealed a small amount of pleural effusion in addition to lymphadenopathy in bilateral cervical, mediastinal and peritoneal areas, and hepatosplenomegaly. A neck lymph node biopsy showed positive expression of EBV-encoded RNA by in situ hybridization, and the presence of serum EBV DNA was confirmed by polymerase chain reaction (PCR). Based on our patient’s clinical course, a diagnosis of EBV-associated infectious mononucleosis, SLE and secondary Sjögren’s syndrome was made, with a SLE Disease Activity Index (SLEDAI) score of 11.
His serum lactate dehydrogenase and alkaline phosphatase levels gradually improved after three weeks of hospitalization, with no treatment. Improvement was also shown on physical examination, and our patient was discharged on day 21.
Two weeks after initial hospitalization, he presented with high fever and mild dyspnea on exertion. Physical examination was unremarkable except for high fever (40°C) with SaO2 of 98% in room air. PCR results for serum EBV DNA were negative and the titers for anti-nuclear antibody (ANA) and anti-dsDNA antibodies declined to low levels, with no clinical signs of SLE exacerbation (SLEDAI score of 3). A chest X-ray revealed interstitial infiltration in both lungs (Figure ), and a chest CT scan showed diffuse, randomly distributed, ill-defined centrilobular micronodules with patchy ground glass opacity in both lung fields (Figure ). Pulmonary function tests revealed impaired gas diffusion with a forced vital capacity (FVC) of 129.2% of predicted values, forced expiratory volume in one second (FEV1) of 100.8% of predicted values, FEV1/FVC of 72.8%, and diffusion capacity of the lung for CO (DLCO) of 66.2% of the predicted value. Fiberoptic bronchoscopy with bronchoalveolar lavage revealed markedly increased cell counts with predominance of lymphocytes (84%) with 1% neutrophils, 1% eosinophils and 14% macrophages. Flow cytometry revealed a predominance of T lymphocytes (99%), with 35.6% CD4+ and 62.8% CD8+ cells; the ratio of CD4+/CD8+ was 0.57.
On the basis of our patient’s medical symptoms, the differential diagnoses included hypersensitivity pneumonitis (HP), EBV-related lymphomatoid granulomatosis, lymphocytic interstitial pneumonia secondary to Sjögren’s syndrome and SLE, and sarcoidosis. To confirm the diagnosis, a lung biopsy was obtained by video-assisted thoracic surgery, and sections from the right middle lobe revealed nodular lesions measuring up to 1 mm in size, containing well-formed, non-necrotizing, epithelioid cell granulomas, comprising lymphocytes, plasma cells, and eosinophils on hematoxylin and eosin staining, whereas peri-bronchiolar cellular infiltration and alveolitis were unremarkable (Figure ). These lesions were randomly located within the central airspaces of the lung, and did not show bronchiolocentric distribution, which is commonly seen in patients with HP. The results of tissue stains and cultures for bacteria, mycobacteria and fungi were all negative, and EBV-encoded RNA expression in the lung was not detected by in situ hybridization. Immunohistochemical staining results for S-100 protein were also negative.
HP was considered as a possible etiology of our patient’s symptoms. However, our patient was not known to have been exposed to any inhaled agents, nor did he show any clinical improvement during hospitalization. In addition, the histopathological features of the lung biopsy were not typical of those of HP. The other possibilities, such as sarcoidosis, pulmonary involvement of Crohn’s disease, SLE, and Sjögren’s syndrome were also considered to be unlikely based on clinical and pathological findings.
Our patient’s condition did not improve: he had continued daily high-grade fever, and there was no improvement in his radiological findings. Thus, prednisolone therapy was started at a dose of 0.5mg/kg/day, as a result of the possibility of specific immunological reactions associated with EBV infection. Our patient showed remarkable clinical and radiological improvement, and he was discharged on a tapering dose of prednisolone.
After one year of follow-up, our patient remained asymptomatic and had no relapse after discontinuation of prednisolone. Physical examination and chest X-ray/CT results were normal. Serological studies showed an absence of ANA and anti-dsDNA antibodies, and the presence of anti-SS-A/Ro antibodies, EBNA IgG, and anti-smooth muscle antigen antibodies.


Our patient was a 52-year-old African-American man who presented to our facility with a two-month history of progressive weakness and numbness in all extremities. A computed tomography (CT) scan and a brain magnetic resonance imaging (MRI) scan revealed a 4.5 × 3.7 × 2.7cm, homogeneous, avidly enhancing mass located in the fourth ventricle, compressing adjacent medulla and cerebellum and extending into the foramen of Luschka (Figure ). Angiography demonstrated multiple dysplastic-appearing branches from the right posterior inferior cerebellar artery supplying the neoplasm.
Our patient underwent a suboccipital craniotomy and resection of the tumor. During the operation, a cavitron ultrasonic surgical aspirator as well as bipolar cautery and suction were used to debulk the tumor, followed by a combination of RHOTON™ dissectors as well as microscissors to separate the tumor. The entire tumor was successfully removed.
Histological examination showed a spindle cell neoplasm with prominent collagenized stroma (Figure A). Mitotic figures were not readily found and the molecular immunology Borstel-1 (MIB-1) proliferation index was low (1%). Meningothelial whorls and psammoma bodies were absent. The neoplastic cells were strongly and diffusely positive for CD34 (Figure B), vimentin and Bcl-2 (Figure A); no immunoreactivity with S-100, CD99 and epithelial membrane antigen (EMA) was detected. A CD31 immunostain highlighted the endothelial cells but not the spindle tumor cells. A moderate reticulin network was demonstrated by reticulin staining, but individual cells were not invested by reticulin fibers (Figure B). The histological features and immunoprofile were consistent with SFT.
Our patient experienced no neurological deficit after surgery, and there were no signs of recurrence at one-year follow-up.


An 11-year-old boy was admitted for biopsy of a skin rash and an enlarged axillary lymph node. No family history of autoimmune disease or autoinflammatory disorder was evident, and he had received no blood transfusion or stem cell transplantation. The patient had suffered from recurrent fever since infancy. At one-month old, the patient presented with fever without source, maculopapular rashes, and poor weight gain. Laboratory examinations revealed elevated levels of liver enzymes and C-reactive protein (CRP). Despite a careful examination, however, neither infection nor neoplasm was detected. There was also no evidence of biliary disorder. As the patient was well fed and his general condition was not much affected, he was followed closely as an outpatient, without therapy. Liver biopsy at seven-month old demonstrates features of chronic active hepatitis, with lymphocyte and plasma cell infiltration in Glisson's capsule yet without fibrosis and with a diffuse hepatocyte ballooning pattern.
Primary immunodeficiencies including phagocyte disorders, antibody deficiencies, complement deficiencies, and combined T- and B-cell immunodeficiencies [] were refuted through further examinations. Liver markers gradually normalized by age 3, whereas CRP levels remained elevated, and skin rash and fever without source relapsed occasionally. This long-standing fever and elevation of CRP levels without infection or neoplasm were considered possible indicators of autoimmune disease. However, as laboratory findings supporting the diagnosis of autoimmune disease such as positive antinuclear antibody test were not obtained, a definite diagnosis could not be established. At age 4, petechiae developed on the trunk, associated with thrombocytopenia and positive anti-dsDNA autoantibodies. Intravenous pulse steroid therapy was administered and this ameliorated both the clinical and laboratory findings. Petechiae and purpura disappeared and remittent fever resolved. Platelet count increased within normal range and CRP turned negative. The elevation of anti-dsDNA autoantibodies diminished within normal limits at this time. Nevertheless, CRP level rose again soon afterwards. Low-dose oral prednisone (5 mg/day) was commenced, but high-dose prednisone was frequently required when high fever developed. Frequent exacerbations and remissions of erythematous rash were observed all the while. At age 7, investigations for short stature revealed growth hormone (GH) deficiency, and GH replacement therapy was started but showed little effect. At age 9, an extensive differential diagnosis was considered for primary immunodeficiencies, especially infantile onset autoinflammatory disorders [, ] with clinical manifestations even if only slightly consistent with those observed in the patient. This involved genetic testing for conditions including cryopyrin-associated periodic syndrome (C1AS1), familial Mediterranean fever (MEFV), tumor necrosis factor receptor-associated periodic syndrome (TNFRSF1A), and hyper-IgD syndrome (MVK). However, none of these genetic defects was identified. Innate immunity deficiencies were excluded as clinical symptoms of this patient were not consistent with those of defects of innate immunity currently known. The similarity of the patient's clinical course and laboratory findings to those of cGVHD suggested the stable long-term persistence of allogenic cells, that is, mMc. Throughout the course, we observed that persistent fever and elevated CRP often resolved, contrary to expectations, when the patient had symptoms of an upper respiratory tract infection. For a definitive diagnosis, a biopsy of the skin and lymph node lesions was planned.
On admission, the patient exhibited markedly short stature (−5.5 SD) unresponsive to GH treatment, hepatosplenomegaly, generalized superficial lymphadenopathy, and urticaria-like rashes. No arthropathy was observed. Laboratory findings revealed anemia (Hb 8.2 g/dL), elevated levels of inflammation markers such as CRP, ESR (79 mm/h), and SAA (563 μg/dL), and hypergammaglobulinemia (IgG 3721 mg/dL). Serum complement levels were normal. Serum levels of IL-6 and B-cell activating factor (BAFF) were elevated at 21.4 pg/L and 3314 pg/mL, respectively. Viral infections such as Epstein-Barr virus, cytomegalovirus, and parvovirus B19 were not identified. Ophthalmologic examination was unremarkable. Thoracoabdominal CT scan showed hepatosplenomegaly and mild systemic lymphadenopathy. Gallium scintigraphy revealed no abnormal accumulation. Bone marrow examination revealed no abnormality. Histological examination of the skin revealed nonspecific inflammation. The perivascular and periappendicular mononuclear infiltration was observed in dermis. That of lymph node showed no increase of plasma cells but of histiocytes and dendritic cells. Epstein-Barr virus DNA and human herpes virus type 8 DNA were not detected.
HLA typing using nested polymerase chain reaction (PCR) with sequence-specific primer typing revealed noninherited maternal antigens below measurable limits (<0.01%) in circulating lymphocytes. On the other hand, female cells were demonstrated in the patient's skin and lymph nodes by fluorescence in situ hybridization labeling for the X and Y chromosomes (). The percentage of female cells, presumed maternal, was 0.2% of all nuclei examined in the skin and 0.3% of those in the lymph node. Since elevated IL-6 was thought to be important in the pathogenesis, tocilizumab therapy was started. After the first tocilizumab infusion, IL-6 level rose further to 2230 pg/L as expected. Fever resolved, and the levels of CRP and BAFF normalized shortly thereafter. IL-6 level decreased but remained elevated (approximately 200 pg/L). The patient's height did not increase significantly, and the skin lesions persisted after more than 100 infusions.


A 66-year-old lady presented to the general surgeons with a history of diarrhoea and abdominal distension. Examination revealed ascites and a CT scan was organised; this showed gross ascites with small pleural effusions, bulky ovaries, a fibroid uterus, and multiple peritoneal and mesenteric deposits. Tumour makers were within normal limits with Ca 125 17, CEA 2.1, and Ca19 9 6.2. A gynaecological oncology opinion was sought and an ascitic drain sited for symptom relief and to obtain possible cytological diagnosis. Histological analysis of ascitic fluid on two separate occasions revealed leukocytes, mesothelial cells, and scattered histiocytes with no malignant cells present. Further immunohistochemistry was performed confirming the cells to be lymphoid in origin.
She continued to be symptomatic with abdominal distension and vomiting and therefore, a diagnostic laparoscopy was performed by the general surgeons. Laparoscopy confirmed gross ascites with what appeared to be widespread carcinomatosis with peritoneal and omental seedlings, bulky uterus and ovaries. Omental biopsies that were taken revealed an inflammatory process suggestive of possible sclerosing peritonitis. Histology showed omental fat with fibrous proliferation at the surface infiltrated by inflammatory cells and nodules of mesothelial proliferation, with no evidence of neoplastic infiltration. Immunohistochemical analysis was negative.
Her clinical condition remained unchanged, and she required further ascitic drains for symptom relief. An MRI scan confirmed CT findings, and her case was discussed in detail at the Gynaecology and Colorectal MDT.
As extensive investigation failed to establish a definitive diagnosis. In view of the small possibility of the fibroid uterus being responsible for her condition the decision was taken to perform a total abdominal hysterectomy, bilateral Salpingo-ophrectomy, omental biopsy, and serosal bowel biopsy. The gynaecological procedure was completed without difficulty with note only of an enlarged fibroid uterus. The peritoneal cavity was generally fibrosed extending throughout the bowel surface. Macroscopically the bowel was firm, solid with no obvious peristalsis due to the serosal fibrosis explaining the symptoms of obstruction. The bowel was inspected by the bowel surgeon and a number of biopsies were taken.
Histological analysis from the peritoneal fluid showed mixed inflammatory cells and markedly reactive mesothelial cells. Interestingly, the ovaries were normal and the uterus contained an infracted ischaemic fibroid. Sections from the omentum and small bowel showed a fibrous process with spindle cells and inflammation. Overall this indicated diffuse sclerosing peritonitis.
Her postoperative recovery was uneventful and at her six-week follow-up she had made an excellent recovery. She remained asymptomatic at her six-month follow-up. A CT that was performed at the time when compared to previous imaging showed complete resolution of her ascites and a normal appearance of the bowel.


A 25-year-old female patient was referred to the Department of Orthodontics, Dental School, Shahed University of Medical Sciences, Tehran, Iran. Past medical history revealed a kidney cyst, but the patient did not suffer from any systemic disease. Clinical examinations eliminated the possibilities of any developmental disorders and syndromes. Extra-oral examination did not reveal any abnormality. In intraoral examination, mild spacing in both of dental arches was noted. Her upper left lateral incisor, second premolar and first molar were in dental crossbite without any functional shift. She had class III occlusal relationships and cephalometric evaluation revealed a class III skeletal pattern (). In her panoramic radiograph, four impacted third molars were present (Figure 2). Significant root dilaceration of maxillary lateral incisors and right first premolar was evident. Surprisingly, an impacted supernumerary canine was noted on the right side of the mandible and a primary canine accompanied by two impacted teeth, both resembling a canine, was present on the left side of the mandible. All of the three impacted teeth had fully-developed roots, and showed the configuration, size and root length of a permanent canine (Figures & ). Occlusal radiograph revealed the impacted tooth on the right was lingual and both of impacted teeth on the left were buccal (). According to the patient, there was no history of tooth extraction. The parents did not have supernumerary teeth; the patient had no siblings.
Fixed orthodontic treatment started for the patient. After six months of treatment including leveling and aligning, the right canine and first premolar were not properly aligned because of the impacted supernumerary canine; therefore, surgical removal of the supernumerary tooth was carried out.
Another surgical session was planned for the left side, in which the distal impacted canine was removed after extraction of the ankylosed primary canine, and a bracket was placed on the mesial canine for forced eruption because of its longer root. Both of the extracted teeth had the size and anatomy of a normal canine ().


A 49-year-old female with active intravenous heroin use was admitted with fever, chills, night sweats, malaise, nausea, and 15 lb weight loss for 2 months. About a month prior to admission, the patient had a foul smelling, draining skin abscess in the left arm at the site of heroin injection, which was treated with a course of trimethoprim/sulfamethoxazole prescribed in the emergency room. Although the abscess resolved, her constitutional symptoms worsened. She reported no cough, vomiting, diarrhea, abdominal pain, or dysuria and was taking no regular medications. She used heroin daily and had a twenty pack-year smoking history. She had been incarcerated for 9 months about five years prior to admission.
On physical examination, the patient was cachectic and malnourished. She was awake, alert, fully oriented and in no acute distress. Temperature was 39.4°C, blood pressure 115/69, heart rate 127, respiratory rate 20, and oxygen saturation 99% on ambient air. The area of the previous injection site infection was healed. Multiple needle track marks in the cubital fossae were noted. There was no cervical and axillary lymphadenopathy, and the lungs were clear to auscultation. Cardiovascular exam showed a regular tachycardia with normal S1 and S2. There were no murmurs or extra heart sounds. Abdominal exam was significant for mild right-upper-quadrant tenderness to deep palpation and a liver span of 14 cm in the right midclavicular line.
Laboratory showed a leukocytosis of 11.2, a markedly elevated serum alkaline phosphatase of 1210 unit/L (normal 38–126) and gamma-glutamyl transpeptidase (GGT) 957 unit/L (normal 12–43), as well as slightly elevated AST 125 unit/L, ALT 55 unit/L, and total bilirubin 1.5 mg/dL with direct bilirubin 0.9 mg/dl. Chest X-ray showed a nodule in the left lower lung. Intravenous contrast-enhanced chest CT demonstrated a peripheral 1.7 cm cavitary nodule in the left lower lobe and a peripheral 3 cm mass-like infiltrate in the right lower lobe (). Abdominal ultrasound demonstrated hepatomegaly without focal abnormality. The patient was empirically started on vancomycin for suspected staphylococcal endocarditis and was placed on respiratory isolation for possible tuberculosis. Since the patient did not produce sputum, bronchoscopy was performed, and an AFB smear as well as bronchoalveolar lavage culture were negative. Transthoracic echocardiogram showed normal systolic function without vegetation. Hepatitis C serology was positive, and HIV was negative. The patient continued to be febrile and tachycardic on day 3, her blood pressure fell to 85/44, and piperacillin/tazobactam was added to vancomycin, along with intravenous fluid resuscitation. Although blood culture was negative initially, gram positive rods, which appeared to be branching, were grown in multiple cultures after 5 days. Nocardia was suspected, therefore, trimethoprim/sulfamethoxazole (TMP/SMX) was started, and vancomycin and piperacillin/tazobactam were continued. Subsequently, the infectious disease consult recommended switching from vancomycin to linezolid to cover adequately for possible disseminated Nocardia infection. Therefore, the patient was placed on linezolid, piperacillin/tazobactam, and TMP/SMX. After the antibiotics regimen was initiated, the patient became afebrile and normotensive and felt much better. On day 14, the patient was discharged home with oral linezolid and TMP/SMX; however, she failed to keep her follow-up appointment and did not take her medications. She was readmitted 14 days later with the recurrence of all prior symptoms.
Approximately 15 days after the first admission, the reference lab reported that the gram positive rods were also acid fast and identified them as Mycobacterium fortuitum. The organism was grown in 6 aerobic bottles of a total of 15 blood cultures obtained during the two hospital admissions. It was sensitive to linezolid, imipenem, ciprofloxacin, amikacin, doxycycline, and sulfamethoxazole; and was resistant to clarithromycin, cefoxitin, and tobramycin. During the second admission, the patient was placed on intravenous linezolid and ciprofloxacin, and oral TMP/SMX with clinical improvement. She underwent transesophageal echocardiogram, which showed aortic and tricuspid valve vegetations (). A liver biopsy showed granulomatous hepatitis (). No organisms could be identified on the liver biopsy culture. However, serum alkaline phosphatase level fell with antibiotic treatment ().
The patient completed a six-week course of intravenous antibiotics and was discharged on oral bactrim and ciprofloxacin with goal of continuing the antibiotics for 6–12 months. She was counseled regarding options for treating her narcotic addiction.


We present a 77-year-old man, with no history of smoking, admitted to the emergency room with a 2 month history of malaise, shortness of breath, and weight loss. His medical history involved controlled heart failure, arterial hypertension, hypercholesterolemia, and obstructive sleep apnea syndrome. Blood count, liver, and renal functions were normal. Chest X-ray showed areas of consolidation in both lung bases, predominantly left.
A CT-scan of the chest demonstrated diffuse bilateral ground glass nodules, ill-defined areas of pulmonary opacities with “crazy-paving” pattern in right lower and middle lobes, and extensive air-space consolidation in left lung (Figures , , , and ). These findings suggested inflammatory or infectious process as first choice, being less likely neoplasic aetiology or organizing pneumonia.
The PET-CT scan (low-dose CT) reported an extensive and heterogeneous deposit of [18F] Fluoro-2-deoxy-D-glucose (18F-FDG) in both lungs with a maximum standardised uptake value (SUV) of 11.30, that correlated with morphological findings described on CT scan. In addition, hypermetabolic lymph nodes were detected in right supraclavicular, left mediastinal, and subcarinal regions (maximum SUV of 6,06). These PET-CT findings suggested as first choice a bilateral inflammatory or infectious process ().
Bronchoscopy demonstrated serous secretions predominantly in the left bronchial tree. Bronchial aspirate, bronchoalveolar lavage, and bronchial biopsy resulted positive for adenocarcinoma. All bacteriological tests performed were negative.
In this case an activating mutation on exon 19 of epidermal growth factor receptor (EGFR) gene was found. Activating EGFR mutations derive in increase of response to EGFR tyrosin kinase inhibitors when comparing with standard chemotherapy [, ]. This finding allowed us to start treatment with the EGFR oral tyrosin kinase inhibitor Gefitinib. Rapid and sustained response was observed in the follow-up CT scan, and the patient remains on Gefitinib for 8 months without evidence of progression (Figures and ) and excellent tolerance.


A 32-year-old woman presented with acute abdominal pain and fever. She had undergone a right radical nephrectomy for pT2N0M0, Fuhrman's grade 2, clear cell renal cell carcinoma 4 months ago. Enhanced abdominal computed tomography (CT) revealed multiple hypovascular tumors in the liver. Laboratory findings were increased white blood cells count, as well as elevations of serum C-reactive protein and lactic dehydrogenase (LDH), which was consisted of subtype 1 and 2 (). Because these laboratory data were similar to those at the diagnosis of primary RCC, we diagnosed multiple liver metastases of RCC. We started 50 mg sunitinib per day for 4 weeks followed by a 2-week rest period as a first line. Sunitinib is an oral TKI. Serum LDH level was 2500 U/L before sunitinib treatment, then transiently decreased to 694. However, it increased up to 8350 U/L again and CT confirmed progression after 3 months (). Sunitinib was stopped, and temsirolimus was administered at a dose of 25 mg per week as a second line. Temsirolimus is an inhibitor of mTOR. Two months after temsirolimus administration, serum LDH level decreased to 233 U/L and CT showed the shrinkage of tumor (). Five months after temsirolimus administration, serum LDH level increased to 2290 U/L again and strongly enhanced tumor's progression was confirmed (). We thought the tumor was switched from hypovascular to hypervascular tumor and rechallenged sunitinib. Three weeks after sunitinib readministration, CT revealed that all tumors turned to be hypovascular tumor again (). Several tumors shrunk and some tumors grew. We rechallenged temsirolimus. One month after temsirolimus rechallenge, serum LDH level decreased to 1199 U/L and CT showed shrinkage of tumors.


A 93-year-old white male presented to the emergency department (ED) with a chief complaint of weakness and recurrent falls combined with a history of osteoporosis, dementia, and an inability to care for himself. Upon examination, decreased breath sounds in the right lung were noted, and a chest X-ray revealed a large right pleural effusion. A computed tomography (CT) scan of the chest confirmed the suspicion of a pleural effusion and revealed multiple rib fractures at various stages of healing, a clavicular fracture, L1-L2 vertebral body compression fractures, and a large lucency at the anterior aspect of the approximate T10 vertebral body (). The patient underwent a thoracentesis yielding 1000 mL of hemorrhagic fluid.
Due to the patient's poor nutritional status and the family's decision to decline further treatment, palliative care was initiated. The patient expired four days after admission. The patient had a history of asbestos exposure in the distant past, and, as a result, the family requested an autopsy to determine whether mesothelioma was the cause of the hemorrhagic pleural effusion.Autopsy showed the body of a well-developed, emaciated adult white male weighing 110 lb and 67.5 inches tall (BMI = 17.0 kg/m2). Examination of the thoracic cavity revealed recent fractures of his left 2nd and 3rd ribs as well as multiple healing fractures bilaterally. No blood was emanating from any of these fractures. Most notably, however, was a 2000 mL haemothorax () which had arisen from fractures of the 10th and 11th thoracic vertebrae with associated laceration of the posteromedial aspect of the right parietal pleural lining, overlying the fracture site (Figures , and ). Adherent blood clots were present near the laceration site as well as on the lung subjacent to the fracture site. The overlying large-caliber vasculature (aorta, vena cava, and branches) was intact and uninjured. Although fibrous pleural plaques consistent with asbestos exposure were observed, no evidence of mesothelioma was present, either grossly or by microscopic examination.
Given the autopsy findings, as well as his history of recurrent falls and underlying osteoporosis, his thoracic spine fractures were most likely sustained from a recent fall and ultimately determined to be the immediate cause of death related to a massive haemothorax. The manner of death was ruled an accident.
A 71-year-old white male status post sigmoid colectomy for removal of a colorectal carcinoma developed acute respiratory distress postoperatively, after being moved from the operating table, and was reintubated. Attempts to wean the patient from the ventilator resulted in continued distress and subsequent respiratory failure. The patient quickly declined and progressed to cardiopulmonary arrest. After approximately an hour of resuscitation efforts and stabilizing the patient, he was transferred to the intensive care unit (ICU), where a chest X-ray revealed a large right pleural effusion of unknown origin. A chest tube was placed and 3000 mL of hemorrhagic pleural fluid was withdrawn. A subsequent chest CT did not reveal an obvious source of hemorrhage (). As time progressed and the patient remained nonresponsive after numerous interventions, it became exceedingly clear that the patient had suffered a severe irreversible anoxic encephalopathy from the prolonged cardiopulmonary arrest. The family agreed to withdraw life support secondary to the patient's irreversible medical condition.
Autopsy showed the body of a well-developed, morbidly obese adult white male weighing 330 lb and 74 inches tall (BMI = 42.4 kg/m2). Examination of the thoracic cavity revealed 1000 mL of serosanguineous fluid with clotted blood within the right chest cavity as well as 400 mL of serosanguineous fluid within the left chest cavity. This fluid originated from the posterior chest wall near the lower thoracic spinal column (). Originally, an aortic injury was suspected, but the aorta was free of acute trauma, demonstrating only focal complicated atherosclerotic plaque (). The hemorrhage arose from a fracture of the 11th thoracic vertebral body (Figures and ), presumably having occurred while moving the patient from the operating table to the hospital bed for transport to the recovery room, as no other sources of trauma could be identified. Microscopic examination of the spine demonstrated an insufficiency fracture secondary to osteoporosis. No evidence of malignancy was present.
Based upon the gross and microscopic findings in the decedent's autopsy, massive haemothorax from a fractured thoracic vertebra with underlying osteoporosis was the cause of death. The manner of death was ruled an accident.


A 60 years old man affected by hypertension, hypercolesterolemia and chronic obstructive pulmonary disease (COPD), went to a hospital with a constrictive chest pain. He was haemodynamically stable and he did not have any signs of myocardial infarction (troponine was negative as electrocardiogram). So a CT angiography was performed, revealing a rupture of an aneurysm of left aortic hemiarch measuring a diameter of 8.8 cm. Due to the pathology and for haemodynamic stability, the patient was sent to our institution. When he arrived he was troubled and suffering. At clinical examination we did not find any pathological signs at the thorax and the abdomen; femoral and peripheral pulses were valid. There were no neurological deficits. At laboratory exams he had moderate anemia (10.2 g/dl), normal creatininemia (0.90 mg/dl) and tachycardia (100 beats) with right bundle branch block at electrocardiogram. A new CT angiography showed the contained rupture of the aneurysm. We evaluated the aneurysm was in zone 2, according to Ishimaru classification [], of the arch, but it would be changed into a zone 1 for a safer deployment of the graft. The study of CT angio showed a proximal neck diameter of 32 mm and a distal landing zone of 28 mm with a short proximal neck that required a surgical debranching. Under general anaesthesia and electroencephalogram monitoring, a debranching of supraortic vessels was performed, avoiding median sternotomy; extranatomic carotid-carotid right to left bypass, with a retropharyngeal course, and a left carotid to subclavian bypass were done, followed by endovascular repair of the aneurysm with deployment of endoprosthesis Relay-BoltonTM 32/155 through femoral percutaneous approach. (Figure ) There were no endoleaks at the completion angiography without any complication in puncture’s site. The patient was referred to Intensive Care Unit (ICU). In the first postoperative day the patient was extubated. He had no nerve injuries and neurological asymptomatic and vital signs were within normal limits. During the 2nd postoperative day he was clinically stable, but a left supraclavicular haematoma had grown up and chest pain was worsen with a diffused pulsatility in the left hemithorax. A CT angiography showed a new rupture of the aneurysm due to a type Ia endoleak for a dislocation of graft (Figure ). The patient was taken to operating room to correct the lesion with deployment of endoprosthesis RelayTM 36/190 with free flow on innominate artery. Again, no endoleaks at the end of the procedure.
The patient was discharged in 17th postoperative day and CT angiography was performed: it showed a complete exclusion of the sac. The CT angiography follow up was strict at 2, 6 and 36 months showing a progressive shrinkage of the sac (now the diameter is 4.63) (Figure ). Echocolordoppler evaluation revealed the normal patency of supraortic bypasses at 36 months.


We present a case of 5 year old girl who presented to paediatric outpatient clinic of R.D. Gardi Medical College and C.R. Gardi Hospital, Surasa, Ujjain, India with complains of decreased appetite since past four to five days, yellowish discoloration of sclera noticed since past two days and deep yellow coloured urine since past two days. Patient had no history of clay coloured stools, fever, vomiting, haematemesis, malena or history suggestive of hepatic encephalopathy. The perinatal history was uneventful with no history of umbilical sepsis. The child had no significant past medical history including that of jaundice. Immunization of the child was appropriate for age. At initial examination her anthropometric measurements showed a weight of 15 Kg and a height of 110 cm. The weight-for-age was between 3rd to 15th percentiles; height-for-age between 50th to 85th percentiles and weight-for-height below 3rd percentile, according to the WHO Reference Growth Standards 2007 ( ). The anthropometric findings were suggestive of acute on chronic malnutrition with stunting. The dietary evaluation revealed a diet inadequate in calories (by one third), proteins (by one fourth) and iron (by half) for her age and sex. The vitals at presentation were heart rate of 120 per minute, respiratory rate of 20 per minute and blood pressure of 98/60 mm of Hg in right in right upper limb in supine position. Four-limb blood pressure measurement was also normal. Icterus was noticed. Examination of ears, nose and throat showed no abnormality. No skin rash was present. No clubbing, cyanosis or oedema of arms or legs was noted. Cardiovascular examination was normal. The lungs were clear on percussion and auscultation. The abdomen was soft with no organomegaly or clinical ascites. A provisional diagnosis of acute viral hepatitis was made based on history, clinical examination and local endemicity of acute viral hepatitis caused by hepatitis A or hepatitis E. Only blood investigation advised was serum bilirubin, which showed total bilirubin 9.29 mg/dl with direct bilirubin of 7.38 mg/dl and indirect bilirubin of 2.41 mg/dl. Patient was treated symptomatically for viral hepatitis and asked to follow up if any danger signs of acute viral hepatitis appear or after 4 weeks. No serological confirmation of viral hepatitis was deemed necessary in view of acute viral hepatitis caused by hepatitis A virus being an endemic disease in the geographic region.
Patient followed four weeks later with persistence of yellowish discoloration of sclera but had no other symptoms. Her clinical examination was unremarkable. A liver profile, complete blood counts and abdominal ultrasonography (USG) were ordered. The complete blood counts were within normal limits and did not reveal any evidence of hypersplenism. The liver profile showed total bilirubin 3.4 mg/dl with direct bilirubin of 3.23 mg/dl and indirect bilirubin of 0.22 mg/dl; aspartate transaminase (AST) 98.2 IU/L (normal values 15–46 IU/L); alanine transaminase (ALT) 109.3 IU/L (normal values 13–69 IU/L); alkaline phosphatase 269.3(normal values 38–126 IU/L); prothombin time (PT) 14 with control of 16; INR 1.2. Abdominal ultrasound showed course ecotexture of the liver with anomalous portal vein. The size of portal vein was 8 mm. The portal vein drained into inferior vena cava. A CT angiography was advised following the above abdominal ultrasound report. The contrast enhanced CT scan of upper abdomen with MIP to include of whole thickness of portal vein revealed that the superior mesenteric vein and the splenic vein join to form a confluence that drained directly in the inferior vena cava. The portal vein drained in the inferior vena cava at the level of head of pancreas caudal to the intrahepatic portion (Figure and ). A dilated inferior vena cava is seen in Figure . The CT scan also showed a mass lesion in right lobe of liver probably an adenoma or focal nodular hyperplasia (Figure ). In view of the above features and absence of portal vein radicals in liver parenchyma a diagnosis of Abernethy malformation type 1 b was made. To complete the work up a fasting blood sugar and serum ammonia was done which were normal. (Blood sugar 71 mg/L and serum ammonia 20 μgm/dL (normal values 10–80 μgm/dL). A skeletal survey did not show any skeletal abnormalities. An echocardiogram with colour doppler did not show any associated congenital cardiac defects.


A 52 year old lady was referred for admission because of increasing severity in depressive symptoms. She had fleeting suicidal thoughts and expressed a belief that a former co-patient was sitting on her brain interfering with her daily activities. She reported a turbulent life history. She was born and raised in a remote community; her childhood remained uneventful and carefree. She completed high school and then took some courses in a trade school. After completing the courses, she started working as a health care support worker for few years, took a break and then resumed after she got married in 1985. Her spouse was quite abusive and subjected her to domestic violence. She remained extremely unhappy with her marital life. She finally got separated from her husband. She has two sons, ages 25 and 20 who live in a different area. Her younger son met an accident and lost his eye sight but is functional and working. The accident and subsequent events were quite stressful for her. She stopped working in the mid of 2011 as her depression and suicidal ideation became disabling. She did not report any substance abuse, legal issues or allergies but had ‘high cholesterol’ problem. She was admitted three times during the year 2011 before this admission. In terms of family history, her mother had committed suicide and sister suffered from depression. She herself had previous history of suicidal attempt. Her current admission was also precipitated following an overdose. Her depression accompanied by delusion of being overpowered was not responding to the previous treatment regime. Upon referral, there was a suggestion for a trial of Electroconvulsive Therapy (ECT). Following admission, her mental status examination revealed severe depression with fleeting suicidal thoughts, delusion of a former male co-patient overpowering her brain with limited insight into her problem. She was initially given a diagnosis of Major depressive disorder with psychotic features.
This diagnosis was based on DSM IV criteria for major depressive disorder. All her blood work and MRI were normal in findings. By definition in terms of phenomenology, delusion is a belief, held with extraordinary conviction, unshakeable, with incomparable subjective certainty and in contrast to person’s socio-cultural background. The patient in this case report stated that a person is overpowering her brain “sitting on my brain”, expressed firm belief of its reality and was unshakeable despite arguments presented against this belief. She was on Mirtazapine45 mg, Clonazepam 0.5 mg twice per day and Quetiapine800 mg per day without any beneficial effect before being referred for admission. Her treatment was reviewed by the multidisciplinary team. The dosage of Mirtazapine was maximised to 60 mg HS, Quetiapine was discontinued and Risperidone was introduced at 1 mg dose. Over the next two weeks, the dosage of Risperidone was titrated up to 4 mg. The patient showed very good response to this combination, her mood improved tremendously, delusions became shaky, gained adequate insight into her problem and dismissed the brain-occupation by the fellow patient as absurd and obsessional. She stated that “The occupation of my brain could be just my thought, it may not be real but it is quite undesirable”. This falls well into the quality of an obsession which is troubling thought, being recognized as once own, cannot get rid of a content of consciousness, realization of it being senseless and persisting without cause. The consideration for ECT was dropped, she was given off unit privileges and overnight passes. A family meeting was arranged in which it was expressed by the family members that she was doing quite well and they were pleased with the improvement.
The current case, presented with major depression with the firm belief that her brain was possessed by a man. She was considered to be suffering from Major depression with psychosis. With change of antipsychotic medication, the intensity of symptoms became less severe; she gained insight into her problem and started to believe that her thought was irrational. The journey appeared to be from delusion to obsession. This is in light with the above conclusions made in the study []. The same author had examined seven cases with features of OCD with psychosis and six of those responded well to addition of antipsychotic or antidepressants with good effect. ECT treatment was deferred in the current case in order to observe the symptoms and effect of prescribed medications. There was no strong evidence available for efficacy of ECT in obsessional disorder. However, one uncontrolled case series reported considerable improvement in such cases year after the treatment although improvement was correlated with improved depression scores []. In the current case, the patient was observed for improvement on the changed antipsychotic medication. A study [] describes co-morbid OCD with schizophrenia. In this, they mention that OCD was associated with more severe depressive symptoms, social dysfunction and worse premorbid functioning. There remains a significant degree of uncertainty and complexity surrounding the issue of distinguishing between delusion and obsessive thought. []
Some authours feel that these symptoms are distinct entities and are distinguishable on the basis of the level of insight a patient displays. []
Apparently, this is a simple case but the intriguing factor was related to the phenomenology. Initially she remained firm about her belief giving a clinical impression of ‘delusion’ fulfilling all criteria of the same. With initiation of changed treatment she gave the picture of an obsession. With multiple interviews, it remained inconclusive whether the initial picture was of delusion or obsession. The case came under debate based on the issue of phenomenology whether the interview approach was inappropriate or the understanding of phenomenology was inadequate. Addition or change of antipsychotic medication can address both issues making the matter more complex. For training residents and clerks, the terms for psychopathology become more pertinent when it is clinically applied. In major depression with psychotic features or depression that is refractory to antidepressant medication, addition of antipsychotics may prove effective. This has been demonstrated in studies. []
In a study, it was concluded that antidepressant-antipsychotic cotreatment was superior to monotherapy with either drug class in the acute treatment of psychotic depression. []
The medical literature does not clearly mention the need for antidepressants in presence of pure psychotic features. In Obsessive-Compulsive Disorder (OCD), Quetiapine augmentation to SSRI’s was suggested to be beneficial in patients with treatment resistant OCD. [] Hence, it is obvious that though (selective serotonin reuptake inhibitor)SSRI is the medication of choice in OCD, addition of an antipsychotic may prove beneficial.
A lesson was however learnt that addition/change of antipsychotic medication may affect mood related disorder, obsession and/or psychosis. This dilemma prompted us to write a case report for the general interest of readers. Though such type of presentations are frequently reported in academic literature, it gives repeated updates about this sort of clinical dilemmas and provide insight into the management issues.


A 59-year-old Greek man was admitted to our hospital nine months prior to the submission of this case report. He was complaining of persistent cough and chest pain that had started one month prior to a medical appointment. A physical examination on admission revealed enlarged left supraclavicular lymph nodes (approximately 3cm) and an asymptomatic firm subcutaneous mass on the dorsum of his penis, close to the glans, just left of the midline. No pain on his penis was mentioned at that time. Laboratory findings showed elevated serum levels of carcinoembryonic antigen (37.3ng/ml), neuron specific enolase (35.2ng/ml), and lactate dehydrogenase (1.590IU/L) and slightly elevated levels of creatinine (1.8mg/dL).
A chest plain radiograph and chest computed tomography (CT) demonstrated a spiculated lung mass (with a maximum diameter of 4cm) on the apical segment of the lower right lobe and multiple metastatic pulmonary nodules, consistent with carcinoma of the lung, as well as enlarged mediastinal lymph nodes (with an aortopulmonary window of 1.5cm and bilateral hilar nodes with a maximum diameter of 2.1cm) and left supraclavicular nodes (of approximately 1cm) (Figure ). A CT scan of the brain revealed no abnormalities, whereas a CT scan of the abdomen showed bilateral adrenal masses, which were larger on the left and were consistent with metastatic disease from lung carcinoma, and enlargements of para-aortic, peripancreatic, and liver portal lymph nodes. The bone scanning and the CT scans of the chest and abdomen revealed osteolytic lesions at the right side of the bodies of T5-T6 thoracic vertebrae and in the posterolateral arches of sixth, seventh, eighth, and ninth ribs, a mass invading the spinal cord, and another mass (with a maximum diameter of 4cm) located next to the ninth right rib.
Magnetic resonance images of the pelvis (Figure ) demonstrated a high-intensity area on T2 and a marginal obscuration of the Buck’s fascia (with dimensions of 2.5×4.5mm), which appears thickened around the left corpus cavernosum. This finding was confirmed by ultrasound imaging of the penis. The patient subsequently underwent a CT-guided percutaneous puncture of the mass in the left adrenal and para-aortic lymph node. A cytological examination confirmed metastatic adenocarcinoma of the lung (strong immunoreactivity against thyroid transcription factor 1, or TTF-1).
A penile malignancy was suspected, and a biopsy of the corpus cavernosum penis was performed. The biopsy led to a histological diagnosis of adenocarcinoma (Figure ). The histology of the specimen was consistent with that of previous lung cancer. Two longitudinal tissue samples from the corpus spongiosum – which measured 1 and 1.8cm, respectively – were obtained for examination. Histology revealed connective tissue with invasion by clusters of cells with marked cellular atypia. The cells showed strong and diffuse immunoreactivity against CK7, CK8, vimentin, and CK34βE12 and focal positivity against TTF-1 and no expression of CK5/6 and CK20 antibodies. The morphologic features and the immunophenotype of the neoplastic cells indicate a poorly differentiated pulmonary carcinoma metastatic to the penile corpus spongiosum (Figure ). Therefore, the patient was considered to have penile metastasis from lung adenocarcinoma.
Radiotherapy was used to treat the metastatic bone lesions three months after the medical appointment. Three weeks later, the patient was referred to us for treatment of lung cancer. He was treated with chemotherapy consisting of carboplatin (area under the curve of 5) and pemetrexed (500mg/m2) given on day 1 in a cycle of three weeks (q21 days). After the third cycle of chemotherapy, restaging of the disease was performed as the clinical performance status was worsening; a chest CT scan confirmed progressive disease with several new lung lesions, but the lymph nodes of the mediastinum as well as the abdominal ones were minimized. Additionally, a CT scan of the brain showed metastatic lesions at the left frontal lobe and the corona radiata. After completion of the total brain irradiation, second-line chemotherapy was administered with paclitaxel (90mg/m2) given weekly on days 1, 8, and 15 (q28 days) for two more cycles. Restaging showed partial response (lung lesions, penile lesions, and left supraclavicular lymph nodes), but bone marrow depression followed.


We present the case of a 30-year-old West African Yoruba man, an indigent Nigerian clergyman, who presented to our facility with recurrent nasal discharge of a history of about one year, recurrent nasal blockage of a history of about five months, and right facial swelling of a history of about one week. The nasal discharge was bilateral, mucopurulent, brownish, foul smelling, associated with excessive sneezing accompanied with intermittent bilateral nasal obstruction five months prior to presentation. The obstruction started from the right nasal cavity and subsequently involved the left side to become bilateral with an associated inability to perceive smell and no history of epistaxis. The right cheek swelling was noticed a week prior to presentation, progressive and associated with facial pain. A nasal examination revealed a pale polypoidal mass in both nasal cavities, characteristic of nasal polyps that do not bleed on touch, with associated mucopurulent sinonasal discharge. No other mass was seen.
A CT scan of the paranasal sinuses showed an isodense lesion in both nasal cavities, maxillary antral and ethmoid, with erosion of the medial and anterior walls of the right maxillary antrum (Figure ).
Our patient had a bilateral nasal polypectomy and intra-nasal inferior meatal antrostomy performed and blackish debris was seen in both the nasal and antral cavities. The antrostomy was widened with a Watson-Williams rasp. The operative findings were a pale glistening mass and black-greyish materials in both nasal and antral cavities, and mucopurulent aspirate from the right antrum.
The surgical specimens were sent for fungal studies and histopathology. Fungal studies yielded trichophyton mentagrophytes and the histopathology result showed inflammatory polyps. Our patient was treated with terbinafine 250mg daily for two weeks, within which time the symptoms subsided. Our patient failed to turn up for follow-up. Our patient presented again four months after surgery with a recurrent right cheek swelling; an X-ray of the paranasal sinuses showed opacification of both maxillary sinuses with erosion of the anterior and medial walls as earlier noted.
Our patient had a bilateral antral washout with warm saline under local anesthesia using the previously made antrostomy. Our patient was then given a trial of flumetasone and clioquinol (Locacorten®-Vioform®; double dilution) intra-antral application after informed consent was obtained. A nasal spray with 4% plain xylocaine was applied to the nasal cavity; flumetasone and clioquinol solution was then applied with our patient in a sitting position and neck slightly extended. An antral cannula was introduced into the maxillary antrum through an old inferior meatal antrostomy (surgically created fistula). Aspiration of the antral content was performed with subsequent introduction of the agent (flumetasone and clioquinol) with a volume of about 5mL into the cavity. An initial pre-application photograph was also obtained from our patient after informed consent was granted. This was repeated serially weekly for six weeks and close follow-up and monitoring of our patient were performed. After the first dose, there was remission of the right cheek swelling, but persistent nasal discharge. After the second dose the nasal discharge stopped with continuous regression of the mass.
A third dose was administered, and our patient successfully presented for the fourth, fifth and sixth dose. An antral aspirate yielded 3.6mL of straw-colored fluid that was sent for cytology and fungal studies. The results of these studies were negative for fungal and bacterial growth. There was improvement in our patient’s symptoms such as nasal clearance and facial pain, which reduced significantly. Our patient has been symptom free for the past four months after administration of the medication intra-antrally.


A 58-years-old male patient reported in the Department of Prosthetic Dentistry, VSPM Dental College and Research Centre, Nagpur with the chief complaint of worn out denture occlusal surfaces and difficulty in mastication (). Detailed medical and dental history revealed that the patient was suffering from neuromuscular disorder (differential diagnosis-trigeminal myoclonus) resulting in repetitive episodes of cyclic, involuntary, and uncoordinated brisk jaw movements. Oral manifestations showed severe wear of occlusal surface of prosthetic teeth (Figures and ). Further it was found that it was his fourth denture set within a span of three years. Although the retention of the denture was satisfactory, he was forced to have his denture remade frequently. This forced the patient to undergo great physical and mental stress of dental procedures and frequent visits to dental operatory. To meet this vague problem and to reduce wear rate of prosthetic denture teeth, it was decided to fabricate the complete denture prostheses with cast metal occlusal surface.
Metal occlusal surface are used to construct denture that oppose natural dentition or for reconstruction of dental arch with metal occlusal surface. Wallace [] used gold for occlusal surface. On comparing the wear of gold, porcelain, heat-cured, and light-cured resin in occlusal contact, Ekfeldt and Oilo [] found that all materials had the greatest loss of substance when the opposing teeth were of porcelain. The heat-cured, unfilled resin was the least wear-resistant material, followed by light-cured resin, porcelain, and gold. The heat-cured resin showed a combined tribochemical and fatigue type of wear. The light-cured resin and porcelain showed mainly a fatigue type of wear, whereas gold showed a combined abrasive and fatigue type of wear. Tanaka et al. [] stated that the traditional denture fabrication technique using resin teeth with metal occlusal surfaces is rather complicated and time consuming. The teeth are arranged on the wax denture and processed in the usual manner. The patient is allowed to use the completed denture for a period of time to harmonize the occlusal surfaces with oral function. The occlusal portions of the teeth are then separated from the base and invested as a resin pattern for casting. The new cast occlusal portions are reset on the base portions with a resin adhesive. Krantz et al. [] described a simplified method for making esthetic cast metal occlusal surfaces. Posterior acrylic resin teeth casted in a nickel-chrome alloy are coated with silane and an esthetic composite resin veneer is applied to the buccal surface. These veneered posterior metal teeth are incorporated in the wax setup and the dentures are processed and finished. Ekfeldt et al. [] conducted a study to compare a gravimetric method and an impression technique in the evaluation of occlusal substance loss. The wear of gold, porcelain, and microfilled resin was studied in vivo. The gravimetric method showed lower substance loss for porcelain than for gold, whereas the microfilled resin had the highest substance loss. The observed findings corroborated with previous findings of the wear mechanism of the materials; that is, gold has mainly abrasive wear in contact with porcelain, whereas porcelain has a fatigue type and microfilled resin a tribochemical type of wear. After complete evaluation of the patient's problem and need, a modified technique of fabricating denture prosthesis with metal occlusal surface was planned. Standard technique using conventional clinical and laboratory steps were followed to fabricate the denture and essential modifications were made to attend anticipated results. Following satisfactory wax try-in appointment, Putty index impression of denture teeth were made for each quadrant separately with polyvinyl siloxane impression material (Aquasil Soft putty/Regular set, Dentsply, Germany). Wax pattern from the same were made using inlay casting wax (Harvard, Blauwachs, Berlin, Germany) with attention to get occlusal and lingual/palatal surface anatomy of each quadrant. Wax patterns were further refined and retentive tags and beads were added for retention of the tooth coloured acrylic facing and also for mechanical locking of the prosthetic tooth to denture base. Patterns were invested in phosphate bonded investment (Bellasun, Bego, Germany) and casted in nickel-chromium alloy (Ruby Dental Products Inc., Osaka, Japan). Castings were finished and polished using standard procedures. Acrylic resin facings were then added to the castings with appropriate tooth coloured heat polymerized acrylic resin (A3) (DPI, Mumbai, India). Acrylic denture teeth were replaced with custom-made metal occlusal surface denture teeth in respective quadrants maintaining the verified jaw relation record on the articulator. Wax try-in was repeated for final evaluation of the jaw relation, phonetics, aesthetics, and occlusion. Prosthesis was processed, finished, and polished following laboratory remount procedure (). Denture insertion was done following the necessary clinical remount procedure (). Recall visits were scheduled after 24 hours, 1 week, 1 month, and then every subsequent 3 months initially. Patient was on regular yearly follow up thereafter. Denture provided the effective services to the patient satisfaction (). Seeing the patient's compliance and complete satisfaction of denture prosthesis, new dentures were fabricated using the same procedure after six years.