A 30-year old, gravida 1, para 0, woman at 24th week of pregnancy presented with right lower quadrant pain, fever and nausea. Axillary body temperature and pulse rate were 37.3oC and 104/min, respectively. Abdominal examination revealed tenderness, guarding, and rebound tenderness at the right side of the uterus. White blood cell count was 19,500 cells/mm3 with 92.8% neutrophils.
The sonographic examination did not reveal any findings consistent with acute appendicitis exception for periceaceal fluid and it showed a normal intrauterine pregnancy. With a preoperative diagnosis of acute appendicitis, a laparotomy was done via a paramedian incision in the right side of the abdomen. Appendix was erythematous and appendicectomy was performed.
The histopathological examination of the appendix revealed acute appendicitis with extensive deciduosis of the appendiceal wall of all the three layers. There were also glandular structures among the decidua which were positive with cytokeratin 7, while the adjacent appendicial mucosa was negative. The diagnosis was endometriosis and decidiosis of appendix and acute appendicitis. The patient was discharged with antibioteraphy without further complaints.
One month later, at 28 weeks’ pregnancy, the patient came again with left lower quadrant pain, high fever and vomiting. Her temperature and pulse rate were 38.5oC and 110/min, respectively. At physical examination, the patient had 7 months of pregnant uterus and acute abdominal findings consisting of mainly left lower quadrant tenderness. Laboratory results showed a white blood cell count of 23, 800 cells/mm3. A pelvic sonogram revealed a normal intrauterine pregnancy at 28 weeks and free fluid between the intestinal structures and a 5 cm complicated left ovarian cystic mass compatible with tubo-ovarian abscess. At laparotomy with a median subumbilical incision, a small amount of purulent free fluid and extensive pelvic deciduosis covering all the pelvic peritoneal surfaces with left tuboovarian abscess was observed. The abscess was drained and left salpingectomy was performed.
Papillary excrescences were observed protruding into the lumen of the mass and ovarian biopsies were obtained. The abdominal cavity was washed with warm saline solution and a drain was placed in the Douglas pouch for drainage which was removed 3 days later. Early postoperative recovery was uneventful but on the 5th postoperative day the patient had uterine contractions and tocolytic therapy was initiated with nifedipine. She gave birth to a 1400 gr healthy male baby with spontaneous vaginal delivery.
Histo-pathological examination revealed ovarian endometriosis with marked decidual changes and hemosiderin-laden macrophages as well as moderate amount of polynuclear leukocytosis in cyst wall. The final pathological report confirmed infected endometrioma with deciduosis. Postoperatively patient received tazocin antibiotherapy for 3 weeks and discharged from the hospital without further complications. At the second postpartum month she was completely recovered and her baby was doing well without any complications related to prematurity.


In August 2012, a 47-year-old, white Caucasian, female patient was referred, by an orthodontist, to the Stomatology Department of the Pontifícia Universidade Católica do Paraná, Brazil, presenting with extreme signs of local pain in the submandibular region and speaking limitations due to hampered mouth opening. Clinically, left submandibular lymphadenopathy was detected, as well as edema in the left side of the floor of the mouth, with a purulent discharge. In addition, a yellowish structure of hard consistency was observed near to the sublingual caruncle. An occlusal radiograph revealed a cylindrical radiopaque sialolith-compatible image, measuring approximately 2 × 1 cm, in the lower left canine region, confirming the diagnosis of submandibular sialolithiasis ().
During the anamnesis, the patient did not report systemic diseases and reported being allergic to Penicillin. Further, the surgical excision was executed in the same day due to the exacerbated symptoms and the favorable position, in which the sialolith took place after a submandibular massage. Under local anesthesia, a 5 mm incision was performed on the mucosa, over the sialolith. In order to move the sialolith close to the incision, the submandibular massage in the posteroanterior direction was repeated. Using forceps, a 20 mm sialolith () was removed from the floor of the mouth. After the sialolith removal, it was possible to see the reinstatement of salivary flow and also the reduction of the purulent discharge. The patient was medicated with Azithromycin 500 mg, Ibuprofen 600 mg, and Paracetamol 750 mg.
In the following week, the patient returned without edema and local inflammation. The submandibular gland function was tested performing local massage, indicating normal salivary flow. Surprisingly, the patient provided a previous panoramic radiograph, dating from March 2011 (), representing the last radiographic exam for conclusion of orthodontic treatment. Despite slight morphological alteration, the orthodontic radiograph allowed for the detection of the sialolith. Based on the lack of early detecting the sialolith on the orthodontic radiograph, a deeper investigation in the patient's dental files was carried out revealing an additional panoramic radiograph, dating from June 2004, in which no sign of sialolith was detected (). Clinical and radiographic followup were performed 6 months after the surgery revealing no alterations.


A 53-year-old Japanese woman was admitted to Niigata Rheumatic Centre, Shibata city, Japan. She had been diagnosed with MS associated with right optic neuritis and thoracic myelitis when she was 25 years old and treated with high-dose prednisolone (PSL). The myelitis had relapsed three times when she was 36, 37 and 40 years old and treated with high-dose PSL. Oligoclonal IgG band was found in cerebral spinal fluid (CSF) and IgG and myelin basic protein in CSF were elevated (4.9 mg/dL and 1.2 mg/dL, respectively). Brain T2 weighted magnetic resonance imaging (MRI) showed high intensity area beside left lateral ventricle indicating asymptomatic plaque lesion due to MS. High intensity area was also shown in T2 weighted MRI of cervical spinal cord. Anti-aquaporin 4 antibody was negative. Slight right hemiparesis remained, and she needed a cane to walk outside. The MS achieved remission and PSL was stopped for 9 years. When she was 50 years old, polyarthritis developed, and rheumatoid factor and C-reactive protein (CRP) levels were high. She was diagnosed with RA. The PSL was restarted at 7.5 mg daily and methotrexate (MTX) was begun. Because the MTX could not be increased over 8 mg/week because of mild elevation of transaminases, tacrolimus (3 mg daily; TAC) was added to MTX and leukocyte apheresis was performed. However, the RA activity remained high: the CRP was 2.3 mg/dL and the disease activity score (DAS28ESR) was 4.94 (moderate disease activity). Furthermore, joint space narrowing of both knees and ankles had progressed obviously over 1 year. Because anti-TNF therapy can exacerbate demyelinating disease, the anti-IL-6 receptor antibody tocilizumab (TCZ) was started at 8 mg/kg every 4 weeks. At the second administration of TCZ, the CRP was <0.1 mg/dL and the DAS28ESR was 2.0 (complete remission). The MTX and TAC were tapered and stopped in 6 months, and the PSL was tapered to 0.5 mg daily in 1 year. The health assessment questionnaire disability index (HAQ DI) in 1 year was 1.88 and functional disability was remained. At the 5-year follow-up, she remained in remission with TCZ.
Serum interferon (IFN) -γ was negative (≤0.1 IU/mL) and serum high sensitivity TNF-α was within normal range (1.6 pg/mL) before starting TCZ therapy. Both of them kept the same levels for a year. Serum IL-6 level was elevated, 51.2 pg/mL (normal range; ≤4.0 pg/mL) before starting TCZ therapy and it was 57.1 pg/mL a year later.


A 62 year-old man who had undergone proctocolectomy and ileal pouch anal anastomosis (IPAA) with a hand-sewn anastomosis for ulcerative colitis eight years previously developed pouchitis that did not respond to topical hydrocortisone (rectal foam) and oral prednisolone 20 mg/day. Prior representative biopsies from the inlet ileum and all thirds of the pouch had not revealed any malignant cells. Four months after the onset of his pouchitis symptoms, which were liquid stools combined with high frequency and incontinence resulting in a erosive dermatitis of the anal region, he was readmitted to our hospital complaining of pain in the anal region, meteorism, lower abdominal pain, diarrhea and loss of weight of about seven kilograms in four weeks.
Physical examination, ultrasonography, laboratory values, and hydro-magnetic resonance imaging (hydro-MRI) revealed no pathological findings except for leukocytosis (13.8 × 109 cells/L). Pouchoscopy of the IPAA showed hemorrhagic and ulcerating pouchitis with stenosis of the proximal pouch (Figures and ). Biopsies showed both acute and chronic ulcerating inflammation with subtotal atrophy of the villi (Heidelberg Pouchitis Activity Score 10, scale 0 to 12, []). Surprisingly, we saw a circumscribed dark lesion in the proximal third of the pouch which we biopsied during a subsequent pouchoscopy. Immunohistochemistry confirmed a diagnosis of malignant melanoma (MM) (melan A positive, S100 and phosphoglucomutase-1 negative, Ki-67 positive in 80% of cells). We then performed thorough staging. A skin check by a dermatologist revealed two benign skin tumors (lentigo simplex and lentiginous compound nevus) that were completely excised. Examination by an otorhinolaryngologist and an esophagogastroduodenoscopy showed no abnormalities. Computed tomography of the thorax and abdomen and MRI of the pelvis showed enlarged regional lymph nodes and a partially thickened IPAA.
Two month after the initial presentation, the patient underwent complete resection of the pouch including the sphincter apparatus. Intraoperatively, the dorsal prostate gland was found to be adherent to the pouch close to the palpable mass. After consultation with our urologist colleagues, a tumor infiltration of the gland was strongly suspected and the prostate was resected en bloc. The patient recovered uneventfully from the procedure.
Histological examination of the pouch revealed ulcerated ileal mucosa and an invasive MM with atypical melanocytes invading the underlying submucosa. The lesion was composed of sheets of epithelioid melanocytes with melanin pigment that were positive for S100 and human melanoma black (HMB)-45 (Figures and ). Six resected mesenteric lymph nodes belonging to the pouch were infiltrated by the same cells but proved to respect the lymph node capsule.
Surprisingly, histological examination of the prostate revealed a synchronous adenocarcinoma (Gleason score 5 (2 + 3), TNM classification pT2, pNx, pMx, L0, V0, R0) although no infiltration by the MM could be evidenced.
In summary, the final diagnoses were MM of the anal mucosa with secondary infiltration of the ileal pouch after restorative proctocolectomy, and adenocarcinoma of the prostate.
All treatment decisions were strictly according to the recommendations of our weekly tumor board. After postoperative interdisciplinary discussion of this patient, close surveillance was recommended since there were no signs of distant metastases and, according to the literature, adjuvant therapy would have no proven benefit in this situation [].
Six weeks after the patient was discharged from our hospital, he was readmitted because of further weight loss, lack of appetite and poor general health. Ultrasonography revealed diffuse metastatic spread to the liver. Multiple percutaneous biopsies taken under ultrasound control showed tumor cells that were negative for S100, slightly positive for HMB-45 and strongly positive for vimentin, confirming metastases from the previously diagnosed MM. We administered palliative chemotherapy with dacarbazine but the patient died of hepatorenal syndrome and subsequent multiorgan failure three weeks later.


A 9-month-old Caucasian female without any birth or past medical history was diagnosed with isolated enophthalmos of the right eye. The mother had noticed a backwards displacement of the right eye 3 months prior to presentation and consulted various specialists who stated a constitutional feature. General examination was normal with no deterioration of health status, neurological, abdominal or skeletal bone integrity. Ophthalmological examination revealed nothing but a mild right enophthalmos. There was no facial disfigurement such as flattening. Pupil size and reactivity to light, direct and consensual accommodation, fixation and following, binocular function, eye-hand coordination, reaction to patching, slit lamp and fundus examination were all normal. Before this isolated enophthalmos, blood and urine samples were collected and orbital computed tomography (CT) undertaken. Routine biology labs, in particular creatinin levels, were normal. An orbital CT scan confirmed the enophthalmos (Figure
a) and showed irregularly shaped osteolysis with periosteal reaction of orbital walls, malar bones and zygomatic arches, predominantly on the right side (Figure
b), arousing suspicion of a tumor. A thoracic- abdominal CT scan showed an 83 mm*43 mm*42 mm retroperitoneal heterogeneous mass located on the left adrenal gland (Figure
) embracing vascular elements and associated with intra-abdominal, left supraclavicular swollen lymph nodes and vertebral condensations, which was evocative of a stage-4 neuroblastoma. The patient was immediately referred to oncology for further investigation and treatment. The stage-4 neuroblastoma was confirmed and the patient treated with chemotherapy and surgical evacuation of residual masses. She achieved complete remission with no recurrence at 10-month follow-up.


A two-and-a-half-year-old female toddler presented with a mass in her right lower leg since one year before. Initially, her parents noticed a pea sized mass growing bigger slowly. The onset was not associated with fever or trauma. The mass grew gradually until the size of a pumpkin on the lateral side of her right lower leg, which caused anxiety to her parents.
The mass was firm in consistency, with no tenderness, noncompressible, and nonpulsating. The overlying skin was glossy. Neurological examination distal from the mass was normal. Range of motion of right knee and ankle was normal (). Radiograph of the right leg showed a soft tissue mass on the posterolateral side. Fibular bone was slightly deformed with periosteal thickening (). Computerized tomography scan revealed that the soft tissue mass was suspected to be rhabdomyosarcoma. FNAB result was benign soft tissue lesion.
Wide excision of the mass was performed similarly to the previous case. Gross pathology of the resected specimen showed an encapsulated, firm, lobulated mass with internal septations. On microscopic view, the tumour consisted of adipose tissue divided into lobules by fibrous septa, myxoid mesenchymal tissue, and lipoblasts. Immature myxoid cells were in the periphery and mature adipocytes in the center; the myxoid area contained lacy network of small blood vessels. The final diagnosis was lipoblastoma ().


An eight-year-old girl presented to our hospital with a painless mass below the left knee. Initially at the age of three years, her parents noticed that she had a mass on her left lower leg. She had undergone radiographic examination and excisional biopsy in other hospital. Several months after surgery, the mass recurred and became bigger. Two months before admission, she returned to the orthopedic surgeon who performed the surgery, and she was eventually referred to our hospital. The mass size was approximately 10 × 10 × 10 cm with smooth overlying skin, was nontender, and had ill-defined margin. There was scar from previous surgery and a fixed equinus deformity of the ankle (). Radiographic examination showed a soft tissue mass in the proximal left lower leg. MRI showed a soft tissue mass with heterogenous signal intensity and ill-defined margin, which infiltrated the surrounding muscles without bone involvement ().
The mass was excised through a posteromedial approach. There was adhesion of the mass to gastrocnemius and soleus muscles, tibialis posterior artery, and tibial nerve. Muscles were conserved except the part of the muscles which was attached to the mass. Microscopically the mass showed lobulated appearance consisted of mature adipocytes and myxoid parts (). Based on clinical manifestations, result of MRI, and histopathology, we concluded it was a lipoblastomatosis.


A 46-yr-old Chinese man was referred to our hospital, with dyspnea and orthopnea. Two months before this, he was admitted to another hospital for suffering from high fever (39.2°C). Large doses of penicillin were intravenously given then because of positive blood culture, which showed Viridans Streptococci. He came to our hospital because of shortness of breath and orthopnea, but no fever for 40 days. Transesophageal echocardiography during operation revealed a saccular structure in the anterior leaflet that bulged into the left atrium throughout the cardiac cycle (Figure ), and also the anterior leaflet of the mitral valve was redundant and prolapsed due to the aneurysm. Severe aortic regurgitation was detected on TEE, which led to the enlargement of left ventricle and mitral annulus. Physical examination found diastolic murmur on the left sternal border and systolic murmur at the apex. Infective endocarditis was suspected, so after admission blood cultures were repeated which were all negative, also medical therapy for heart failure was started and resulted in clinical stabilization rapidly.
The patient underwent aortic valve replacement with 23 mm bileaflet mechanical prostheses (St. Jude Medical) and mitral valve plasty with autologous pericardium. At operation, no vegetation was found on the coronary cusp, the aortic valve appeared to be thicken and coarse, but no vegetation. A cystic cavity of 15 mm on the anterior mitral leaflet was clearly visualized from the left atrium and was unbroken (Figure (A)). The mitral valve leaflet was smooth and no visible evidence of endocarditis was found. Therefore we just resect the MVA at its bottom and the aortic valve as normal. After resection of the MVA, an oval deficit of 6 mm in diameter on the anterior mitral leaflet was visualized (Figure (B)), which was repaired with autologous pericardium (10 × 10 mm) as usual, and the patch was placed at the left ventricle side of the valve through left atrium (Figure (C)). The autologous pericardium patch was excised, treated with 0.625% glutaraldehyde solution for 10 minutes, and rinsed with physiologic saline. Also a 30 mm C Type Sorin prosthetic ring was used for mitral annuloplasty due to the enlargement of the mitral annulus (Figure (D)). Transesophageal echocardiography during operation showed mild regurgitation. After operation, although cultures of the removed aortic leaf-lets and MVA were negative and further pathology showed no evidence of inflammatory infiltrate, which was due to the administration of enough doses of antibiotics before the operation, vancomycin was still intravenously given to prevent the recurrence of infection (1.0 g q12 h for 2 weeks). The patient recovered uneventfully and was discharged asymptomatic on the tenth postoperative day. 3 months later transthoracic echocardiography still showed a perfect result.


A 22-year-old female was admitted to the Jilin Cancer Hospital (Changchun, China) in May 2013. In January 2009, a 2.0×3.0 cm painless mass was surgically removed from the left parotid gland of the patient. Postoperative pathology characterized the mass as a lymphocytic hyperplasia; however, parotid duct cells were also detected within the lymphocytes. Morphological results and immunohistochemical staining supported the diagnosis of a BLEL.
After six months, the left parotid gland became locally swollen again and intermittent management with traditional Chinese medicine was unable to achieve remission. In October 2010, the size of the mass reached 5.0×6.0 cm. A computerized tomography (CT) scan of the parotid gland indicated a soft tissue mass in the left neck area without uniform density or a clear border with the left parotid gland, and evident infiltration in the surrounding tissue. The mass was considered to be a recurrence of the BLEL in the left parotid gland; however, the patient did not agree to further surgical therapy. The mass in the left parotid gland area became gradually enlarged, and was more evident when the patient became ill with a cold; however, the mass was slightly reduced following anti-infective treatment (mainly with cephalosporins). The patient also received traditional Chinese medicine for six months and local acupuncture therapy for 40 days. However, the mass in the left parotid gland area was continually increasing in size and reached a diameter of 15 cm. On April 27 2013, the patient developed a fever of up to 40°C, accompanied with shivering, weakness and a loss of appetite, which was more severe in the afternoon and at night. Remission was observed following self-medication with oral ibuprofen; however, the temperature of the patient increased after several hours and a light yellow, clear liquid was discharged from the ulceration on the skin surface of the left parotid gland mass. On May 4 2013, the patient visited the Jilin Cancer Hospital, where the local ulceration and infection of the left parotid gland mass was managed with anti-infective treatment for three days; however, the high temperature of the patient did not decrease and the mass did not reduce in size. The patient was admitted to the hospital for further therapy on May 8 2013, and physical examination identified a large mass of 17×14 cm on the left side of the patient’s face, a red and swollen skin surface and multiple ruptures in the middle of the mass with a light yellow liquid discharge. The peripheral skin of the mass was tenacious and swollen, and the skin temperature was high. A blood routine examination and liver and renal functions were found to be normal. Color ultrasound revealed a visible 18.2×8.5-cm low-echo area with strong-echo stripes under the skin on the left side of the patient’s face, without the appearance of a clear margin. Color Doppler flow imaging revealed an abundant blood flow in the mass and the absence of normal parotid gland tissue, indicating a solid space-occupying lesion on the left parotid gland. A CT scan of the left parotid gland revealed an abnormal mass in a low-density image and an undefined margin of the lesion, with the superior border extending towards the orbit, the inferior border at the submaxillary level, the inner border at the left parapharyngeal space and the outside border at the skin of the left-side of the face. The mass reached 13.4×9.7 cm in an axial view, showing uniform density and a CT value of 47 HU (), with the left parotid gland unable to be distinguished. The lesion was wrapped around the left sphenoid bone and mandible. In addition, morphological changes were observed, indicating recurrence of the BLEL in the left parotid gland with bone resorption of the sphenoid bone and mandible. A bacterial culture of the mass discharge indicated a Staphylococcus aureus infection, which was sensitive to levofloxacin. The body temperature of the patient returned to normal levels following the administration of levofloxacin for one week. A biopsy was not performed due to the weakness, high fever, local swelling and infection of the patient, as well as the abundant blood flow detected in the mass with color ultrasound. No consensus was reached with regard to further surgical intervention. Drugs inhibiting vascular endothelial growth (Endostar and Shenyi capsules) were administered for five days, but were discontinued by the patient due to local pain and swelling. Chemoradiotherapy was subsequently considered; however, due to the young age of the patient and the examination results not indicating malignance, the patient did not agree to the therapy.
Following failed conservative treatment, DC-CIK therapy was initiated based on the immune index result of the patient, which found the percentage of inhibitory T cells to be 32.2% (CD8+CD28−; normal range, 9.8–21.8%) and the CD4+/CD8+ ratio to be 0.42 (), indicating low immunity. The patient signed informed consent for the treatment protocol, which was approved for tumor patients by the Health Department of Jilin Province (Changchun, China) and the Ethics Committee of the Jilin Cancer Hospital.
Antigen-presenting DCs and CIK cells were produced using heat shock protein-70, extracted from a head and neck squamous carcinoma cell strain supplied by the Jilin Cancer Hospital, and cocultured with cord blood DCs.
The duration of the first treatment session was 10 days, after which the treatment was discontinued for 10 days. Following the initial treatment course, the lesion in the left parotid gland area was reduced, while the local infection and skin ulceration were soothed. Biotherapy was continued for three months (four sessions of treatment), after which the left parotid gland lesion disappeared and the skin ulceration was healed.
Parotid gland CT scans () indicated that the primary lesion of the left parotid gland area disappeared gradually and the bone destruction was completely recovered. The patient was discharged from hospital and the condition remained stable without signs of recurrence during a follow-up period of 10 months; however, the patient remains under close observation.


The current study presents the case of a 71-year-old male patient who was diagnosed with RCC in September 2011. At that time, the disease was at an advanced stage. The primary tumor in the lower pool of the right kidney was infiltrating the adjacent structures and the patient exhibited synchronous mediastinal and pleural metastases, with the latter causing persistent pleural effusion and markedly declining lung function. Due to the poor performance status and risk of side effects, the patient refused to initiate the disease-controlling sunitinib treatment and chose to proceed to the optimum supportive care. The patient presented with subcutaneous metastases to the lower lip and back of the neck 11 months after the diagnosis. The patient received palliative radiotherapy (split course, 15/5 Gy) to the rapidly growing lower lip metastasis. The tumor diameter was 1.5 cm when the treatment was initiated. However, no clinical response to radiotherapy was obtained, and three weeks following the treatment the tumor had more than tripled in diameter. Thus, the patient was evaluated at the Department of Oral and Maxillofacial Diseases (Helsinki University; Helsinki, Finland). At the time of admission the patient had a spontaneously bleeding mass (size, 60×60 mm) in the lower lip and the anterior mandible area (). In addition to this, there was a group of smaller subcutaneous metastases located at the subcutaneous nuchal area, which did not exhibit symptoms. Resection of the lip metastasis was performed with 5-mm clinical margins and for this reason, the resection was extended to the bony surface of the mandible. The lower lip was also partially resected as the small subcutaneous metastases had continued to spread into the lip mucosa (). To prevent wound tension following closure, the skin was dissected subcutaneously from the resection line to the upper neck, pulled over the chin to cover visible bone, and resuspended with transcutaneous sutures to the titanium plate (MatrixMFACE Plating System; Synthes Holding AG, Solothurn, Switzerland) in the mandible (). The patient was satisfied with the outcome at the three-week postoperative follow-up and no clinical sign of recurrence was observed (). Histological examination via immunohistochemical staining () identified the tumor as metastatic RCC and the mass was resected with clear lateral margins.


A 75-year-old male patient admitted to emergency service because of swelling and redness on the left eye and wound discharge under the eye. It was found out that a skin incision occurred at 2 cm below the left eye with razor blade 2 days ago. Redness, swelling, and efflux developed in this incision region within 2 days progressively. Patient specified that the periorbital region crusted over. In medical history of the patient, no additional disease or a condition related with immunosuppression was observed except having bypass due to coronary artery disease. In the physical examination, left periorbital area was hyperemic and edematous and temperature was increased in this area. Under the left eyelid, an irregular 2 cm skin lesion with discharge and necrosis was observed. There was a widespread edema at the left side of the face. There was a minimal periorbital edema at the right site (). High fever was present as a pathological vital finding. Patient's otorhinolaryngological examination was normal otherwise. A neutrophil dominant leukocytosis (15.000/mkrl) was observed in CBC. Sedimentation, 78 mm, and C-reactive protein (CRP), 393 mg/L, were detected. Biochemical parameters were normal. In the ophthalmologic examination, loss of sight was present in the left eye (0/1 snellen). In the anterior segment examination chemosis, periorbital edema, and echimosis were seen. Chorioretinal atrophy was detected in the dilated fungus assessment. In the orbital magnetic resonance examination, rise in the density of periorbital adipose tissue was observed. After taking swab culture sample, patient was started on parenteral Vancomycin + Ampicillin-sulbactam treatment. It was observed that necrosis spread within hours (). Since the clinical situation continued to progress, an emergent deep surgical debridement was performed. Pathology specimens were taken. Wound site care was made by the application of pomades with topical antibiotics and topical rifampicin. Following the debridement, it was observed that periorbital edema began to regress prominently on the 1st day of the treatment. Treatment was carried on with daily wound care and parenteral antibiotherapy. Wound site culture resulted as positive for Streptococcus pyogenes on the 3rd day of the treatment. Patient's antibiotherapy was rearranged as stopping the vancomycin therapy and continuing 1.5 g parenteral ampicillin sulbactam therapy (4 times/day) based on the consultation of department of infectious disease. In the 3rd week, wound healing speeded up and debrided areas got epithelized. The patient was discharged from the hospital with slightly cosmetic defect. In the 1st month, periorbital fasciitis completely disappeared. Unfortunately, the patient failed to attend for follow-up.


A 62-year-old Caucasian male with history of chronic obstructive pulmonary disease, gastroesophageal reflux disease, and knee osteoarthritis presented with seven months history of gradual, dull, and constant abdominal pain affecting the right lower quadrant (RLQ). The pain radiated to his right groin and down his anterior right thigh. Patient also reported 10 months history of mild but persistent epigastric discomfort, dysphagia, and odynophagia with solids and liquids. The patient denied history of fever or chills but did report significant weight loss (>10 lb) and night sweats over the last six weeks. The patient had an extensive tobacco history of two packs-per-day for fifty years, as well as an extensive alcohol history despite abstinence the preceding months. Three months prior to admission to our service, patient underwent laparoscopic cholecystectomy due to the abdominal pain that was described above. However, his abdominal pain persisted postoperatively. Patient presented to our emergency department and was sent home on oral antibiotics for iliopsoas phlegmon as per CT findings thought to be secondary to infection. He presented for the second time two weeks later complaining of severe abdominal pain; repeat CT scan of abdomen and pelvis did not show any significant change in the iliopsoas phlegmon. Vital signs were within normal. Physical exam showed a cachectic man that appeared older than stated age. Abdomen was soft, nondistended and with moderate tenderness to palpation in RLQ and suprapubic area with no evidence of peritoneal inflammation. There were normal bowel sounds and no hepatosplenomegaly. The rest of the physical exam was normal. Initial laboratory results on admission revealed hemoglobin to be 12.6 g/L and otherwise normal CBC. Renal function was within normal. Total protein was 5.8 g/L, albumin was 2.9 g/L, alkaline phosphatase was 1619 mg/dL, ALT was 51 UI, AST was 43 UI, GGT was >1200 u/L, C-reactive protein was 3.7 mg/dL (normal range <5 mg/dL), and ESR was 52. Blood cultures were negative. An abdominal ultrasound reported normal liver measuring 15 cm, no definitive intrahepatic biliary dilatation and mild extrahepatic biliary dilation (CBD measuring 10 mm) that was likely related to cholecystectomy, suggested as not significant. Ultrasound was not done to evaluate psoas muscle. A repeated CT scan of abdomen-pelvis reported ill-defined areas of low density involving the right psoas muscle from origin to insertion and inflammatory changes along the right psoas muscle and right iliacus muscle (Figures and ). Soft tissue lumbar MRI showed persistent thickening and heterogeneity of the entirety of the right iliopsoas musculature. Axial T1 post contrast MRI (images of the lumbar spine) showed normal appearing muscle segments on the left side. Significantly enlarged and enhancing right psoas and paraspinal musculature on the right side (). The differential diagnoses suggested by the radiologist were infectious process versus neoplastic infiltration. Given the malignant suspicious of the lesion, using CT guidance, a fine needle aspiration biopsy of the right psoas mass was performed. The core biopsy sections revealed atypical cells with high nuclear cytoplasmic ratio and hyperchromasia in an infiltrative pattern and were positive for cytokeratin immunostain confirming the diagnosis of metastatic poorly differentiated carcinoma (). CT of head, neck, and chest was done because of significant smoking history, however, it was negative for any abnormality suggestive of malignancy. Given progressive dysphagia, patient underwent upper endoscopy, which revealed esophageal stricture that was dilated. Stomach showed no endoscopic evidence of ulcers or masses. Upon reaching the duodenum, ERCP was attempted for elevated alkaline phosphatase and GGT using regular scope but cannulation was not achieved; near the ampulla, a protruding luminal mass was seen in the second portion of the duodenum (). Duodenal biopsy was taken which showed similar tumor cells in an infiltrative pattern consistent with poorly differentiated adenocarcinoma (). Upon discussing the results with the patient, he refused any type of therapy and requested palliative care. He was discharged to hospice care facility.


A 74-year-old woman was admitted to the dermatology outpatient clinic of our hospital with complaints of deformity and thickening of the toenails that had continued for nearly three months. In the dermatological examination, of bilateral toenails, subungual hyperkeratosis in varying degrees, yellow-brown discoloration, and onycholysis were observed (). The general physical examination was normal. Chronic diseases were absent, with the exception of hypertension. The patient did not have chronic or familial genetic diseases that could have caused nail disorders, malignancy, or previous trauma. The patient revealed that she had traveled to the Far East within the previous six months. The patient reported no use of systemic corticosteroid or broad-spectrum antibiotics. Other immunosuppressive conditions associated with Rhodotorula infection, such as AIDS, were absent. The patient's toenail samples were sent to the microbiology laboratory for fungal culture.
Clinically suspected of onychomycosis, according to nail culture results, the patient was diagnosed with DLSO caused by R. glutinis.
Antifungal therapy was initiated with 400 mg/day oral itraconazole; seven-day pulse therapy was planned at intervals of three weeks. The clinical evaluation of the patient before the start of the third cure, clinical recovery was detected. The patient's treatment is still underway; a fungal culture was planned again after six treatments.
The toenail samples were cultured on 2 Sabouraud Dextrose Agars (SDA; Salubris, Turkey) in the microbiology laboratory. One of SDA was incubated at 37°C and the other was incubated at 25°C. SDA, which was incubated at room temperature for four days, was observed to be a pinkish-orange pigmented colony (). The pure passage of growing colonies was performed on the SDA medium (). The Gram staining of these colonies was observed in the yeast cells forming blastospores. The yeast was thought to be Rhodotorula, due to its orange-pink pigmented appearance. The urease test was performed. The two strains of C. albicans (American Type Culture Collection (ATCC) 10231 and ATCC 24433) were used as a negative control, and Cryptococcus neoformans (ATCC 24067) was used as the positive control. Urease activity was positive.
The tested pathogen was indicated as Rhodotorula glutinis/mucilaginosa according to the Vitek automated identification system (bioMérieux, France) using Yeast Biochemical Card 2 (YCB). The species identification of the strains was performed at the Public Health Institution of Turkey-Mycology Reference Laboratory (PHIT-MRL). The pinkish-orange color, mucoid-appearing yeast colonies on SDA at 25°C, the morphological evaluation in the Corn Meal-Tween 80 agar, observed oval/round budding yeast at 25°C for 72 hours, no pseudohyphae, determination of positive urease activity at 25°C for four days, assimilation features detected by API ID 32C (bioMérieux, France) kit, evaluated together with conventional mycological methods identified the species as Rhodotorula glutinis [].
Susceptibility tests of the strain to amphotericin B, fluconazole, itraconazole, voriconazole, posaconazole, anidulafungin, and caspofungin were performed using the microdilution method (M27-A3), recommended CLSI in PHIT-MRL. Quality control (QC) was performed using Candida parapsilosis ATCC 22019 and Candida krusei ATCC 6258. Due to the fact that after 24 hours of incubation there was no bacterial growth in growth control well and poor growth after 48 hours, minimum inhibitory concentration (MIC) was determined, after a 72-hour incubation according to the CLSI-M27A3 recommended resistance limit values []. Antifungal susceptibility test results were as follows: amphotericin B (MIC = 0.5 μg/mL), fluconazole (MIC = 128 μg/mL), itraconazole (MIC = 0.125 μg/mL), voriconazole (MIC = 1 μg/mL), posaconazole (MIC = 0.5 μg/mL), anidulafungin (MIC = 0.5 μg/mL), and caspofungin (MIC = 16 μg/mL).


The patient was a 46-year-old woman with a history of myasthenia gravis who presented with chief complaints of fever and general malaise. The patient was diagnosed as having RA in 2006, and she was treated with methotrexate (MTX). Because of insufficient efficacy, adalimumab (ADA) was added in 2010. Because of secondary failure, ADA was switched to golimumab (GLM), beginning at 50 mg/4 weeks in 2012. Although the disease activity remained high even after the dosage of GLM was increased to 100 mg/4 weeks, tacrolimus (TAC) was administered concomitantly. During these periods, the RA disease activity ranged from moderate to high. The patient visited our hospital in June 2013 due to a fever and general malaise. Vital signs at presentation were temperature 38.6°C, blood pressure 113/70 mmHg, pulse 87/min, and SpO2 97% (room air). Physical findings included lower abdominal tenderness and rebound tenderness.
Blood tests showed a WBC count of 15,300/μL and CRP of 13.25 mg/dL, indicating an elevated inflammatory response. Table lists the results of the blood tests at presentation.
Abdominal computed tomography (CT) images showed an intrauterine foreign body and ascites. High-density peritoneal adipose tissue and peritoneal thickening were also observed (Figure ).
Based on a gynecological examination, the patient was diagnosed as having pelvic peritonitis due to an intrauterine infection associated with a contraceptive ring and an ascending infection. The contraceptive ring was removed (Figure ). The patient was on anti-rheumatic drugs, which were all discontinued. She was started on an antimicrobial agent, flomoxef (FMOX), at 2 g/day. The FMOX dosage was increased to 3 g/day from the 3rd day of disease and cont for 10 days. Her fever disappeared from the 4th day, and her inflammatory response then gradually decreased. No exacerbation of symptoms occurred even after the FMOX treatment was stopped, and the patient was discharged on the 14th day.
Streptococcus agalactiae (group G beta-hemolytic streptococcus) and Streptococcus constellatus were cultured from the removed contraceptive ring, while blood cultures were negative.
Following the onset of the peritonitis, the joint swelling and tenderness improved during treatment. However, the RA symptoms relapsed 2 months after the onset of the peritonitis. Therefore, administration of GLM + MTX + TAC was re-started at that time, after which there were no signs of infection.


The first patient was a 6-year-old female who at 6 weeks of life had a surgically resected cystic lymphangioma of the sigmoid colon (with primary colorectal anastomosis and appendectomy). She was previously well; however, she began experiencing symptoms of intermittent periumbilical abdominal pain, accompanied by vomiting, constipation, and abdominal distension, which necessitated two separate admissions. On the first admission, she was treated for acute intestinal obstruction, which spontaneously resolved after several days at the hospital. Three months later, she presented with periumbilical pain for two days followed by vomiting, abdominal distension, and headaches. On examination, her abdomen was noted to be mildly distended, but no discrete masses were palpated. She had no family history of malignancy or congenital malformations. On this admission, her haemoglobin (Hb) was found to be 5 gm/dL and she required transfusion of 250 mLs of packed red cells. Her posttransfusion Hb increased to 9.5 gm/dL. Abdominal X-ray did not show any bowel dilation or air fluid levels. Given the patient's past surgical history, there was a high index of suspicion for recurrence of the intra-abdominal lymphangioma. Other differentials included intestinal obstruction and mesenteric adenitis. A computed tomography (CT) scan of the abdomen with IV (intravenous) contrast was performed which showed a cystic mass on the colon and she had surgical intervention. Findings of the surgery included a retroperitoneal cystic lymphangioma enclosing a 50 mL organized clot in the left iliac fossa, multiple peritoneal adhesions, mesenteric windows, adhesions enclosing small bowel into artificial sac, and a ventral incisional hernia. Histology confirmed cystic lymphangioma. The patient has been followed up with regular ultrasound scans (USS) for the past 9 years and there has been no evidence of recurrence or intestinal obstruction thus far.


A 4-year-old male presented with 2-day history of periumbilical pain and a past history of occasional constipation without any significant past medical history and no family history of malignancy or congenital abnormalities. On examination he was found to have a mildly distended, nontender abdomen with diffuse left flank firmness, but no discrete mass was appreciated. The patient continued to have colicky abdominal pain but no constipation or vomiting. CT scan abdomen with IV contrast was performed which showed a 30 cm by 10 cm left sided isodense retroperitoneal mass attached to the lower pole of left kidney (). Based on imaging, the possible differential diagnoses included neuroblastoma and cystic lymphangioma. The patient underwent an exploratory laparotomy which revealed a 20 cm by 30 cm multiloculated cystic mass arising retroperitoneally from the coeliac plexus of lymph. The hilum of the cyst was anterior to splenic artery displacing pancreas laterally to the right. The histology of the specimen confirmed intra-abdominal cystic lymphangioma. Yearly follow-up with USS for the past 8 years has shown no recurrence thus far.


The patient was a 4-year-old female who was referred from a rural health center with 2-week history of progressive abdominal distension. She was otherwise asymptomatic with a positive history of passing stool and flatus. There was no significant past medical history or history of prior abdominal surgery or trauma. She had no family history of malignancy or congenital abnormalities. On abdominal examination, a mass arising from the pelvis was palpable. An abdominal USS revealed a 14 cm by 7 cm by 12 cm fluid filled structure in the left half of the abdomen extending into the pelvic cavity (). Abdominal CT with IV contrast showed a 13 cm by 8 cm by 12 cm cystic abdominal mass that appeared to arise from the pelvis extending into the abdomen and displacing the bowel bilaterally (). Two days subsequent to admission, the patient had exploratory laparotomy and cystectomy. A 15 cm by 12 cm mesenteric cyst containing chocolate coloured fluid was found at the splenic hilum surrounded by a jejunal loop and bordered by the tail of the pancreas. She had uneventful recovery with no evidence of recurrence to date. Histology showed a mixture of lymph vessels and smooth muscle, features suggestive of lymphangioma ().


The patient was an 8-year-old female who presented with a 4-day history of a swelling to the left side of her chest that was increasing in size and she was otherwise asymptomatic. She had no medical problems and no significant family history. Interestingly, she was known to have a lymph node in the same area 7 years prior and was scheduled for surgical excision, but it resolved and hence no surgery was done at that time. On examination, she was found to have a 3 cm by 3 cm firm, smooth mass in her left axilla with no surrounding lymphadenopathy. USS of the chest showed a heterogeneous solid and cystic mass in the left axilla (). The patient had needle aspiration which revealed a bloody aspirate and this was followed by a course of oral antibiotics; however, the swelling failed to resolve. CT scan of the chest with contrast done subsequently demonstrated a left axillary cystic mass with no intrathoracic extension (). She had complete surgical excision of the cystic mass. However, the proximity of the lesion to developing breast tissue posed a challenge to surgical resection. Follow-up in clinic for the past 3 years has shown symmetrical development of breasts with no deformity to the left breast. Histology confirmed the diagnosis of lymphangioma.


A 13 year old female living in a remote rural area came to our clinic with an 8 year history of deformities in the extremities which had gradually became worse till she was unable to walk. The patient over the years had developed recurrent fractures in her legs and arms after minor falls. The family was poor and lived in a remote area far away from proper medical facilities. She was treated by local healers for her fractures which resulted in bowing of legs and arms. There were no gastrointestinal symptoms of abdominal pain or diarrhea. She was brought to the clinic carried by her father. The patient used her arms to drag herself around the house and developed fractures followed by severe bowing of her arms. She had been diagnosed with rickets and iron deficiency anemia by doctors in community hospitals and had received Vitamin D and iron supplements many times without improvement. She had never had a complete workup to find out the cause of her rickets. The patient also had failure to thrive. On examination the patient was pale, weight was 11 kg and height 97 cm (below the 3rd percentile for her age) (Figure ). She had severe bowing of her arms and legs.
Due to the presence of anemia, failure to thrive and rickets the initial impression was malabsorption probably due to celiac disease and the laboratory work up was done accordingly.
Initial laboratory investigations that were done are mentioned in Additional file : Table S1. X-rays of her upper and lower limbs showed diffuse osteopenia and bowing of both legs and forearms with blurring of the metaphyseal lines. It also showed dense transverse lines in tibia and ulna suggestive of looser’s zones indicative of severe rickets (Figures and ).Anti- endomysial antibodies titer was 80 (normal is negative), anti-tissue transglutaminase IgA was positive 75 U/ml (normal <2.5 U/ml) and anti-tissue transglutaminase IgG was negative. Upper endoscopy was done with small intestinal biopsy. The duodenum showed scalloping and fissuring of the small bowel suggestive of celiac disease. The histopathology report of the small intestine showed severe villous atrophy grade IV with crypt hyperplasia consistent with celiac disease. Old Marsh-Oberhuber classification: Type 3c: Total villous atrophy with completely flat mucosa and increased intraepithelial lymphocytes (Figure ).
The patient was started on a gluten free diet. She also was started on Vitamin D supplements, calcium and iron replacement. The patient returned for follow-up 5 months later, she was feeling better, her weight had increased to 14 kg and her height had increased to 97 cm. She had taken iron and calcium supplements for a very short period but she did continue on a gluten free diet. The family was very poor and on further questioning on their dietary habit, it lacked many of the main constituents and was very low in vitamin D for most of her life but she did live in a very sunny area and before becoming crippled she had adequate sun exposure. Her diet before diagnosis was found to consist mainly of grains and breads with little protein and after being diagnosed and receiving instructions on a gluten free diet it was changed to rice and potatoes. Laboratory investigations showed some improvement from 5 months earlier these are shown in Additional file : Table S1. The patient was seen by an orthopedic surgeon who wanted the general condition of the patient to improve before considering any surgery.
The patient and her family were further instructed again on a gluten free diet emphasizing the available options. She was given further iron, calcium and Vitamin D replacement, the patient was not able to come back for follow up but 4 months later the family phoned and said she had markedly improved and had started walking.


A 70 year-old man with history of myeloproliferative disorder presented with 3 years of balance problems and falls. He felt he was veering to one side when he was walking. He denied tremors and memory problems. On examination, he scored a 24/30 on the Folstein Mini Mental Status Exam (MMSE) (Folstein et al., ) with deficits in attention, recent memory, and executive function. He had dysmetric saccades. Sensory exam was notable for inconsistent vibratory loss in the feet and temperature loss in all extremities. He had mildly increased tone in both arms and mild kinetic tremor when drawing spirals. He had mild bradykinesia bilaterally. He had no evidence of dysmetria. His gait was wide based and ataxic. He was profoundly unstable when standing or walking, with almost immediate falling. He refused to use a walking aid. Brain MRI showed cerebral volume loss, hyperintensity in the cerebral peduncles, and the MCP sign. He was admitted due to a lack of safety with his balance and had a three-week rehabilitation stay. There he developed a sudden onset bilateral leg weakness. Workup revealed a large soft tissue mass in his spinal cord extending from T1 to T9 causing significant cord compression. It was resected and pathology was consistent with extramedullary hematopoeisis. He then underwent successful radiation therapy. After continued rehabilitation, he was able to regain prior function and is currently walking with a walker. His family history was remarkable for a sister and multiple grandchildren with FXS. His mother had fragile X-associated primary ovarian insufficiency (FXPOI). His FMR1 CGG repeat size was 99. He met criteria for definite FXTAS, with tremor, ataxia, and the MCP sign on MRI.


A 75 year-old woman presented with a one-year history of balance problems, which she described as wobbling when she walked. She had fallen once. She denied tremors, but had decreased hearing in both ears, urinary frequency, and fatigue. Her past history was remarkable for diabetes, end stage renal disease, and vitiligo. On examination, she had normal cognition, saccadic pursuits, and transient endgaze nystagmus. She had increased tone in the right arm without cogwheel rigidity, anterocollis, and mild kinetic tremor with handwriting. Reflexes were absent in the extremities and sensation was decreased to temperature. Dysdiadochokinesia was present in the left hand and bradykinesia in the left leg. She was unable to stand or walk in tandem and got off balance when she turned quickly. There was no retropulsion on the pull test. Brain MRI showed cerebral and cerebellar volume loss with white matter hyperintensities in the periventricular region.
The following year, she had a fall and had surgery for a “pinched nerve” in the neck. She began to have visual hallucinations in the hospital and was started on haloperidol. Her examination had dramatically worsened: she was wheelchair bound and was no longer able to walk unless she had assistance. Her MMSE was 18/30. She then developed delusions and depression. Quetiapine and venlafaxine were added and haloperidol discontinued. She received aggressive inpatient and outpatient rehabilitation over the next 3 months and regained the ability to walk using a walker. Her FMR1 CGG repeat sizes were 90 and 29. She had a daughter, two nephews, and one niece with FXS. She met criteria for definite FXTAS, with tremor, ataxia, and white matter disease on MRI.


A 72 year-old woman presented with progressive difficulty walking since 1998. She initially attributed this difficulty to pain in her feet, and was subsequently diagnosed with plantar fasciitis. By 2000–2001, she developed postural dizziness and soon developed a slow, festinating gait with decreased arm swing as well as fatigue, cognitive slowing, and trouble sleeping. By 2009 she was wheelchair bound. MRI of her brain revealed white matter lesions in the pons and periventricular regions. Testing for autoimmune disorders and spinocereballar ataxias (SCA) was negative. She failed treatment with amantadine and was started on carbidopa/levodopa 25–100 mg twice daily and her walking improved.
On examination at age 75, her MMSE was 25/30, with deficits in executive function, recent memory and verbal fluency. She had apraxia in her left hand and foot, a positive glabellar reflex, and trouble mimicking on the left side. She had absent vertical optokinetic nystagmus, slowed vertical saccades, and square wave jerks. She was bradykinetic and had resting tremor in both arms. Her gait was remarkable for short stride length, frequent freezing, difficulty with tandem gait and impaired postural reflexes. Her carbidopa-levodopa was weaned due to persistent nausea. Donepezil 10 mg daily was started given her cognitive decline. She continued frequent physical therapy at home, but her symptoms continued to progress. She is a FMR1 premutation carrier with 88 FMR1 CGG repeats. Her son has intellectual disability but has not been tested for FXS. Her father had severe tremor and ataxia. Her sister has fragile X-associated premature ovarian insufficiency (FXPOI). She has a male cousin with FXTAS and three female cousins who are also premutation carriers. Based on her MRI as well as ataxia and tremor, she met criteria for definite FXTAS.


An 80 year-old man presented with a three-year history of worsening falls. He had developed shuffling gait, a soft voice, was choking on food, and had a masked facial expression. He denied tremor. He had been diagnosed by a prior neurologist with Parkinson disease and started on 3.5 tablets of 25–100 mg carbidopa/levodopa. By report, his symptoms did not improve. On examination at the age of 80, his MMSE was 28/30. He had a positive glabellar reflex and negative applause sign. He had impaired vertical gaze, impaired vertical optokinetic nystagmus, and slowed saccades. He had symmetrically increased tone in both upper extremities and bradykinesia in all four extremities. He had left sided shoulder elevation with a mild rightward head turn. He had mild rest tremor in the left hand and mild kinetic tremor when drawing spirals. There was no evidence of dysmetria. He had a positive pull test, was unable to perform tandem gait, and took multiple steps to turn. His steps were slow and short. He was unable to have a MRI, but brain CT showed ventriculomegaly. He was started on 5 mg twice daily of memantine given complaints of poor memory, however this was subsequently discontinued as it proved ineffective. His falls initially decreased in frequency after physical therapy. Within 8 months of presentation, he was unable to walk on his own, even with a walker. He had a retrial of carbidopa/levodopa 25–100 mg three pills daily. His bradykinesia improved mildly, but he began experiencing delusions and hallucinations. The psychosis improved and entacapone 200 mg three times daily was added. The patients' family felt the entacapone helped the speech problems, but not the other motor features. He was a FMR1 premutation carrier with 68 FMR1 CGG repeats. His grandson had FXS. He met criteria for probable FXTAS given his tremor and ataxia, but lack of a MRI.


A 60-year old male patient presented with the
complaint of hematuria. Thinking as bladder tumor
by cystoscopic examination, transurethral
resection (TUR) was performed. Through histopathological
findings, he was diagnosed with noninvasive
low grade transitional cell carcinoma (Ta
TCC). He was given an eight-week course of intravesical
mitomycin C. During his follow-up care,
urine cytology was beneficial due to the following
factors: atypical cells with nuclear enlargement,
wrinkled nuclear membranes, and little hyperchromasia
nuclei, distinct or multiple nucleoli. Furthermore,
there were eosinophils nearby the atypical
cells in the material (). Suspicious urinary
cytology was reported. The cystoscoy detected a
hyperemic area seen at the contact of the anterior
wall and the dome. Punch biopsy results from that
area identified the following factors: low degree
pleomorphism , abnormal nuclear morphology and
disordered orientation of the urothelium. In some
area, surface epithelium was detached, but in some
other areas, there was superficial maturation of the
epithelium. Based on histological and cytological
findings, these cytomorphologic changes were due
to adjuvant therapy, mitomycin C, applied for the
patient.
This report aimed at reminding the cytomorphologic
changes of mitomycin C may be misinterpreted
as carcinoma (in situ), so review of the literature
and presence of eosinophiluria are required
for a proper identification of the drug-induced
changes.


A 53-year-old man presented to the Nephrology department with generalized malaise and decreased urine volume for 5 days. He denied nausea, vomiting or fever. The medical history included hypertension for 10 years and type 2 diabetes mellitus for 8 years, both of which had been well controlled. One week before the admission, he underwent laparoscopic cholecystectomy (LC) for right upper quadrant abdominal pain, which had been diagnosed as cholelithiasis and cholecystitis. The laboratory data pre- and post-op are summarized in Table
.
At the current admission, physical examination showed a chronically ill-appearing man in no acute distress, with stable vital signs. The patient was awake, alert and oriented. There was no increase in jugular venous pressure. Pulmonary and cardiac examinations were unremarkable. Abdominal examination showed well healed small scars left by the LC. Mild bilateral pretibial pitting edema was present in both lower limbs. Neurological examination was unremarkable.
The results of an electrocardiogram and a chest/abdominal CT were unremarkable. An ultrasound showed relatively enlarged kidney size (right, 11.1 × 5.1 cm; left, 11.8 × 5.9 cm). Further investigations including complete blood count (CBC), serum creatinine (SCr), urea nitrogen (BUN), electrolytes, liver function, serum uric acid (UA), electrolytes and serum biomarkers. Laboratory results summarized in Table
 indicated elevated SCr, hyperuricemia, hyperphosphatemia, hypocalcemia as well as severe anemia and a slightly decreased WBC count. The remaining of lab tests, including serum tumor biomarkers were normal. The patient’s clinical manifestations of AKI and electrolyte disturbances were consistent with acute TLS.
After admission, the patient was transfused and intermittently hemodialysed, after which SCr and uric acid (UA) decreased gradually, with diminution of edema. Two weeks later, SCr had almost returned to normal and dialysis was stopped. Changes of serum biochemical parameters are shown in Figure
. Since the anemia continued and there were no signs of solid tumors, we highly suspected hematological malignancies and performed several bone marrow examinations. Bone marrow smears from the first two showed no signs of malignancies. There was mild dyshaematopoiesis on the third examination but was not possible to make a definite diagnosis. The biopsy was normal and the molecular analysis returned negative.
The patient visited the clinic every 2-3 months after discharge. Investigations showed HGB was fluctuating in the range of 7.5-9.0 g/dL while SCr and UA remained normal. Chest/abdominal CTs were done 3 months and 9 months later with unremarkable findings. However, the patient complained of persistent progressively worsening malaise. Eleven months later, he was admitted again because of refractory anemia. At this time, bone marrow smear indicated erythroid hyperplasia and significant dyshaematopoiesis of all three lineages. The major loss of the D20S108 signal suggesting 20q- along with minor gains of D8Z2 signal was discovered by fluorescence in situ hybridization (FISH), which was consistent with a G banding karyotype. MDS was confirmed and the patient was classified as low-moderate risk group, hence no chemotherapy. He has been on regular clinic follow-up and the condition has not deteriorated.
The most commonly used diagnostic laboratory and clinical criteria for TLS are those proposed by Cairo and Bishop
[]. A clinical diagnosis of TLS includes one clinical symptom and two laboratory criteria, and at least two laboratory criteria must be present for three days before treatment or up to seven days after treatment. In our case, the patient fulfilled one clinical symptom (acute renal failure) and two laboratory criteria (hyperphosphatemia, hyperuricemia), thus meeting the clinical diagnostic criteria for TLS. In the absence of precipitating chemotherapy or radiotherapy, we considered this as a STLS case.
Acute TLS is usually observed after the initiation of cytotoxic chemotherapy of malignancies, especially hematological malignancies with a high turnover rate. The severity of TLS depends on tumor burden, tumor type, proliferation rate, baseline uric acid level and the sensitivity of the tumor to chemotherapeutic agents
[]. STLS prior to the initiation of chemotherapy is rare, especially in the absence of evidence of a tumor, including hematological malignancies and solid tumor. According to previous studies, STLS most commonly occurrs in Burkitt lymphoma and non T-cell acute lymphoblastic leukemia
[], but it has also been reported in acute lymphoblastic leukemia (ALL)
[], acute myeloid leukemia(AML)
[], myelofibrosis
[], metastatic germ cell tumor
[] and solid tumors
[].
To the best of our knowledge, our case is the first STLS reported in association with MDS. The initial presentations of significant hyperuricemia, hyperphosphatemia, metabolic acidosis, AKI and relative hypocalcemia were all consistent with STLS. In 2003, Yang et al.
[] reported a 32-year-old man who had been diagnosed as MDS with refractory anemia and excess blasts in transformation subtype developed TLS after a single 1.0 g dose of methylprednisolone. However, in our case, the patient did not fulfill the diagnostic criteria for MDS at the onset of STLS. Instead, he was diagnosed as MDS almost one year after the episode of STLS. When the STLS occurred, repeated bone marrow cytology and biopsy did not show evidence of a large tumor burden or of tumor types which had been reported in association with STLS. The etiology in our case is inconclusive. Potential causes for STLS include endogenous secretion of glucocorticoid with infection
[] and fever
[]. In Daisuke et al.’s case of STLS in ALL
[], it was believed the STLS might have been triggered by a febrile gastroenteritis-like illness one month prior to the first admission and accelerated by the concomitant urinary tract infection. In our case, the patient had experienced abdominal pain consistent with cholecystitis before the admission and it had been only one weeks after a surgery when the AKI developed. The endogenous secretion of glucocorticoid triggered by the prior infection and the stress of the operation could be considered to be causes of the STLS.


A 73-year-old female patient was referred to our department with abdominal pain, swelling, constipation, nausea, and vomiting for 2 days. She had hypertension for 10 years, chronic obstructive pulmonary disease for 6 years, and type 2 diabetes mellitus for 4 years in her medical history. She had no history of abdominal surgery, trauma, jaundice, rectal bleeding, and weight loss. On physical examination, abdominal distension, tinkling, and increased bowel sounds were observed. There was generalised abdominal tenderness, guarding was positive, and rebound was negative. The ampulla was found to be empty on digital rectal examination. In laboratory tests, the leukocyte count was 12,500 K/μL (normal range: 4.6–10.2 K/μL), and the blood glucose level was determined to be 150 mg/dL. Other biochemical tests were within normal limits. Air-fluid levels localised in the upper left quadrant were determined by abdominal X-ray. Ultrasonography could not be performed because of dense intestinal gas. On abdominal tomography, small intestinal segments were observed to be dilated in the left quadrant and distal part of ileum; colon segments were collapsed (). The patient underwent surgery with the diagnosis of acute intestinal obstruction. There was no free abdominal fluid at the exploration. The colon was observed in the normal position with lower right localisation of the caecum. The small intestines were palpated under the gastrocolic ligament, which was then opened. The intestines were located in the lesser sac surrounded by a sac, which was opened (). The intestines were in a dilated position, and intestinal perfusion was normal. No space-occupying lesion was found. The terminal ileum had entered into the right lower part of the transverse mesocolon (right side of the middle colic artery) and was obstructed there. No cohesiveness or input-output section similar to herniation was found in this transition area. This area was similar to the right localisation ligament of Treitz. After the obstruction, the ileum moved 4 cm further and was joined to the caecum (). It is defined as intraperitoneal hernia form of intestinal malrotation []. No other small intestinal part in the abdominal cavity except this 4 cm ileum segment was noted. There was no possibility of internal herniation at the point where the terminal ileum passed through the transverse mesocolon. Therefore, it was thought that congenital malrotation caused an obstruction in this case. The obstruction was opened by widening the hole through which the ileum passed in the transverse mesocolon. The intestines were pulled from this aperture to the normal position in the abdominal cavity. The defect in the mesocolon was covered with sutures. The patient started to ingest food orally on the third postoperative day, and she was discharged uneventfully on the fifth day. There was no complaint at the first-month follow-up. The complaint of swelling that had occurred repeatedly for the last 2 years also disappeared.


A 36-year-old male presented to the Department of Oncology of Changzhou Tumor Hospital (Changzhou, China) with a persistent cough, which he had suffered with for two years, 1-day hemoptysis and shortness of breath. The patient was currently smoking 100 packets of cigarettes per year and had an otherwise unremarkable medical history.
A physical examination revealed decreased breath sounds in both lung fields, however no other signs were revealed. Positron emission tomography/computed tomography (CT) scans revealed a mass measuring 6.3×6.2 cm in the right middle lung lobe, and the maximum standardized uptake value (SUVmax) was measured as 9.4, with atelectasis of the right lower lobe. There were multiple ground-glass opacities (GGOs) in both lungs, the largest of which was positioned in the right apicoposterior segmental lung lobe, which measured ~2.0×2.5 cm, and the SUVmax was 2.4. The supraclavicular lymph nodes were grossly enlarged on both sides. There was no metastasis observed in any other organ.
A fiber-optic bronchoscopy detected a stenotic lesion at the opening of the right middle lobe bronchus. A bronchoscopic biopsy confirmed the diagnosis of adenocarcinoma (). Genotype analysis, using an amplification refractory mutations system, determined that the patient had expression of wild-type epidermal growth factor (EGFR), a commonly mutated oncogene. FISH and IHC showed that the patient was negative for the echinoderm microtubule-associated protein-like 4 (EML4)-anaplastic lymphoma kinase (ALK) fusion protein. The patient’s clinical stage was determined as T4(IpsiNod) N3M1a (contralateral lung) (stage IV).
One cycle of chemotherapy was administered, containing pemetrexed (500 mg/m2, d1) and cisplatin (75 mg / m2, d1). One month later, the patient’s disease had progressed; the shortness of breath had worsened, and the pulmonary stenosis (PS) had deteriorated from 1 to 3. The CT scans showed that the targeted lesion in the right middle lobe had shrunk,; however, the nodules in the left lung had become enlarged, and there was a new lesion measuring ~1.0×1.3 cm at the hilus of the left lung. The presence of GGOs in both lungs had increased and enlarged (). The ROS1 protein was detected using IHC, with a staining intensity of >70% 2+ or 3+ () (). Furthermore, a break-apart FISH procedure was used to test for ROS1 gene rearrangements; however, the result was negative when the cutoff value was set at 15% () ().
The patient had severe PS and was being treated with no other targeted therapies, therefore crizotinib treatment was orally administered at the standard dose of 250 mg twice daily. One week later, the patient’s symptoms had improved and the PS was measured at 1, a decrease as compared with the previous measurement. Following four weeks of crizotinib treatment, the CT scans detected an improvement; the targeted lesions in the right lung and left lung hilus had shrunk and the GGOs had almost disappeared (). According to the Response Evaluation Criteria in Solid Tumors criteria, version 1.1 (), the patient’s cancer had elicited a partial response to the crizotinib treatment.


In September 2012, a seven-year-old male was admitted to the Department of Pediatric Surgery, University of Siena (Siena, Italy) with a feminization syndrome, which presented as bilateral gynecomastia. Increased somatic growth was observed (bone age was advanced by more than two years) and a unilateral adrenal mass was diagnosed by ultrasound (USS). The symptoms had begun approximately eight months prior to admission, however, the patient had received treatment for an endocrinological disease. The patient’s plasma testosterone was lower than normal (0.57 ng/ml, normal range, 2.7–10.9 ng/ml) and the androstenedione level was within the normal range (3.08 ng/ml, normal range, 0.4–3.1 ng/ml). The plasma estradiol level (54.6 pg/ml, normal range, <50 pg/ml) was greater than normal. The presence of the mass was confirmed by magnetic resonance imaging (MRI), which revealed a unilateral lesion of the adrenal gland, measuring ~3.8×2.7×3.2 mm. The lesion was well-defined and no additional abdominal abnormalities were present. Furthermore, no lymphadenopathies were observed. The results for metanephrines and catecholamines obtained from a 24-h urine test were negative. In addition, a computed tomography scan revealed that there was no involvement of the chest. Surgical excision of the mass was performed via an open anterior transperitoneal approach and the entire adrenal gland was removed (). A biopsy was performed to remove three lymph nodes, as recommended by TREP guidelines (), however, they did not appear to be grossly involved. The histological analysis identified that the lymph nodes were not involved and determined that the mass was an ACN, with a low risk of malignity. There were no complications during or post-surgery, therefore, the postoperative care was normal and the patient was discharged on postoperative day six. The initial follow-up was conducted one month following surgery, and an abdominal USS and hormone profile were performed. The abdominal USS was normal and the hormone profile revealed reduced levels of plasma testosterone (<0.1 mg/ml, normal range, 2.7–10.9 ng/ml), androstenedione (<0.3 ng/ml, normal range, 0.4–3.1 ng/ml) and estradiol (<20.0 pg/ml, normal range, <50 pg/ml) levels. However, the cortisol level (23.6 pg/ml, normal range, 10–52 pg/ml) was normal. The next surgical follow-ups are expected to be two and six months following surgery.


A 25-year-old male presented to the Qingdao Municipal Hospital (Shandong, China) with a two-month history of painless swelling on the right side of the face. The medical history of the patient was unremarkable. On clinical examination, a large swelling was identified on the right side of the face that was hard and bony on palpitation. A CT scan revealed a giant, multilocular and hypodense lesion of ~6.5×5.0 cm in size, with a well-defined margin (). The lesion involved the majority of the right maxillary bone, maxillary sinus and zygomatic bone. The third molar tooth was completely impacted (). Based on these observations, a tentative diagnosis of a cyst was proposed.
Under general anesthesia, the lesion was enucleated as a whole, using the Caldwell-Luc approach. The tooth that was associated with the lesion was simultaneously removed to avoid tumor fragmentation. Histological examination during the procedure identified a cystic tumor. The wall of the cyst was lined with a stratified squamous epithelium and a corrugated keratinized lining (). A diagnosis of KCOT was determined. To eliminate the possibility of residual cystic tissue, the margin surrounding the lesion was ground and then frozen with liquid nitrogen three times. Prior to the freezing of the surgical site, the adjacent tissues were protected using dry gauze pads. Subsequently, the patient exhibited a good recovery and no recurrence has been observed within the eight-month follow-up duration.


In November 2012, a 65-year-old male was admitted to the First Affiliated Hospital of Chongqing Medical University (Chongqing, China) with dizziness that had persisted for one month and described the occurrence of melena for two weeks. The physical examination was notable for pallor and rashes, and the patient had developed erythematous and maculopapular skin lesions on the legs and hands (). The patient refused to undergo a skin biopsy. Maculopapular rashes appeared around the injection site minutes after intravenous injection. Blood tests were performed and the results were as follows: Hemoglobin level, 6.4 g/dl; hematocrit, 19.10%; platelet count, 58×109/l; and total leukocyte count, 4.55×109/l, with 8% blast cells and 6% mature basophils. The fecal occult blood test was positive, the level of ferritin was 1,138 ng/ml and the levels of vitamin B12 and folic acid were normal. Serum autoantibodies, including the anti-neutrophil cytoplasmic antibodies (ANCA), myeloperoxidase (MPO)-ANCA and proteinase 3-ANCA, were negative. A gastroscopy demonstrated a gastric antral ulcer (stage, A1), according to Sakita’s endoscopic staging system () and the endoscopy biopsy revealed mild mucosal inflammation and no specific features. ().
The results of a bone marrow aspirate (stain, Wright-Giemsa) were as follows: Blast cells, 13%; atypical immature basophils, 14%; mature basophils, 8%; normal mature neutrophils, 11%; and erythroid cells, 13%. The blast cells contained basophilic granules, and the vacuoles in the cytoplasm and nuclei were oval and bilobed and contained nucleoli (). The blast cells were positive for peroxidase and periodic acid-Schiff staining (PAS) and negative for α-naphthyl acetate esterase. The blast cells exhibited metachromasia as a result of staining with toluidine blue. Six days later, the bone marrow aspirate with Wright-Giemsa stain was conducted again, and showed 13% blast cells, 16% atypical immature basophils and 8% mature basophils, with 9% normal mature neutrophils. Cytogenetic studies using GTG banding revealed an abnormal karyotype, 44 XY,add()(p26),-5,-11,add()(q32),+mar1, +mar2,inc[18]/46,XY[2] (). Flow cytometric analysis of the blast cells was positive for cluster of differentiation (CD)11b, CD13, CD22, CD25, CD33 and CD123 (). BCR-ABL fusion was identified to be negative by reverse transcription-polymerase chain reaction analysis and no c-kit D816V mutation was detected.
Following the above-mentioned assessments, the diagnosis of ABL was determined. Induction therapy with daunorubicin (45 mg/m2 daily) intravenously for three days and cytarabine (100 mg/m2 daily) intravenously via continuous infusion for seven days was initiated at the First Affiliated Hospital of Chongqing Medical University (Chongqing, China) on day 13. The hemoglobin level was 5.7 g/dl; the hematocrit was 17.10%; the platelet count was 40×109/l; and the total leukocyte count was 4.41×109/l, with 4% blast cells and 4% basophils. Loratadine, cimetidine and steroids were introduced into the treatment regimen due to concerns regarding the patient’s increased histamine levels and the rashes and melena subsequently disappeared. The patient’s bone marrow examination on day 21 was hypocellular with 1% blast cells, 6% atypical immature basophils and 19% mature basophils, with 24% normal mature neutrophils. The hemoglobin level was 7.4 g/dl; the hematocrit was 22.80%; the platelet count was 31×109/l; and the total leukocyte count was 2.95×109/l, with no blast cells and 12.2% mature basophils. The rashes reappeared, therefore, reinduction therapy was initiated with idarubicin (8 mg/m2 daily) intravenously for three days and cytarabine (100 mg/m2 daily) intravenously via continuous infusion for seven days. Three weeks later, the bone marrow aspirate (stain, Wright-Giemsa) showed 3% blast cells, 8% atypical immature basophils and no mature basophils, with 58% normal mature neutrophils. The hemoglobin level was 6.9 g/dl; the hematocrit was 21.00%; the platelet count was 34×109/l; and the total leukocyte count was 3.71×109/l, with no blast cells and 0.8% mature basophils. The patient was discharged from hospital. However, one month later, the patient contracted a severe pneumonia infection and succumbed as a result of a multiorgan system failure without evidence of relapse.


A 70-year-old man with a history of hypertension was referred to our emergency department with a severe retrosternal and upper abdominal pain that started after he had been vomiting several hours before presentation. At admission, he was diaphoretic and in respiratory distress. Blood pressure was 210/100 mmHg, pulse rate 95 beats/min, oxygen saturation was 95% and core temperature was 36.1°C. Physical examination revealed extensive cervical and thoracic subcutaneous emphysema but was otherwise unremarkable. Laboratory results were normal by the time of presentation. A computed tomography (CT) scan revealed a rupture in the left distal part of the oesophagus, a pneumomediastinum and left-sided pleural effusions (Figure ). Conservative treatment, with cessation of oral intake, nasogastric suction, administration of intravenous fluids and parenteral nutrition, intravenous broad-spectrum antibiotics, proton pump inhibitors and drainage of the pleural effusion by left-sided thoracostomy was initiated in the ICU. After 5 days, however, he developed a fever. Follow-up CT scan demonstrated severe mediastinal contamination and left-sided loculated pleural empyema (Figure ). Open thoracic surgery was performed with debridement and drainage of the mediastinum and the pleural cavity, after which he made a slow but full recovery.


A 52-year-old man visited our clinic with symptoms of foreign body sensation and puffiness in his right eye, which had persisted for 4 months. He was originally diagnosed with allergic conjunctivitis by a general ophthalmologist and was treated with olopatadine eye drops (Pataday; Alcon, USA) once daily and 1% prednisolone eye drops (Pred-Forte; Allergan, USA) four times per day for 3 weeks. He was referred to our clinic after the treatment was unsuccessful, even after oral prednisolone 30 mg/day for 3 days with a tapering schedule was added to topical therapy for an additional one week.
Upon referral to our clinic, the patient stated that his symptoms were negligible when he woke up in the morning, but gradually increased over several hours and persisted with no fluctuations during the day time. There was no itching, pain, or other symptoms in his right eye and no specific symptoms in his left eye. His medical history included hypertension and hyperlipidemia, for which he had been treated for the previous five months with once daily amlodipine besylate 5 mg/atorvastatin 10 mg (Caduet; Pfizer Inc, USA) and once daily candesartan cilexetil 8 mg (Atacand; AstraZeneca, UK), respectively. He reported no history of other systemic diseases, drug allergy, atopic disease or trauma, but he reported always sleeping on his right side. Physical examination revealed no other specific findings such as pitting edema.
Slit-lamp examination showed moderate conjunctival chemosis in his right eye. Swelling was gravity dependent, starting in the interpalpebral area and gradually increasing toward the inferior half, leaving a normal superior bulbar conjunctiva (Figure A and B). The bulbar conjunctiva showed no evidence of a dilated lymphatic channel, and the palpebral conjunctiva showed no papillary reaction or follicular hypertrophy. Both eyes showed no evidence of conjunctival hyperemia or injection, and his left eye was normal. Vision was 20/20 in each eye and intraocular pressure, measured by Goldmann applanation tonometry, was 16 mmHg in his right eye and 18 mmHg in his left eye. Exophthalmometric measure was normal, and neuroimaging using magnetic resonance imaging with contrast enhancement showed no abnormalities such as an orbital mass or venous or lymphatic obstruction. Scheimpflug imaging revealed marked swelling of the inferior bulbar conjunctiva in the right eye, but the anterior chamber depths of the two eyes did not differ significantly (Figure C and D).
Serum IgE and antibody titers for grass and house dust mites were normal. Blood tests, including renal and thyroid function tests, protein concentration, inflammatory markers such as erythrocyte sedimentation rate and C reactive protein, complete blood count and serum chemistry, showed results within normal ranges. The patient had not received any other topical or systemic treatment since the referral from the general ophthalmologist. After the patient was referred to the department of medicine for evaluation of allergic and other systemic diseases, his antihypertensive medication was changed to telmisartan 40 mg/hydrochlorothiazide 12.5 mg (Micardis Plus; Boehringer Ingelheim, Germany). Slit-lamp examination 1 month later showed an almost complete absence of conjunctival chemosis, and he reported that his symptoms were much improved. Therefore, the patient was followed up without any ocular treatment. All of his symptoms had completely remitted and remained stable at his last follow-up visit, 8 months after the change in systemic medication (Figure ).


A 70-year-old Japanese man underwent a medical checkup in March 2005, which showed an abnormal prostate specific antigen (PSA) value (4.8 ng/mL), and he consulted a urologist. He underwent needle biopsy of the prostate. The histopathological findings indicated an adenocarcinoma with a Gleason score of 3 + 3. The number of positive cores was 4 out of 8 cores (right lobe: 1/4, left lobe: 3/4). The digital rectal examination showed no abnormal findings. The clinical stage was T1cN0M0. In 2006, he underwent a low-dose-rate brachytherapy (LDR-brachytherapy) using a preplanning method and modified peripheral loading at our hospital. The prescribed dose was 145 Gy. Regarding dosimetric parameters using CT images 1 month after seed implantation, the minimal dose (Gy) received by 90% of the prostate gland (D90) was 183 Gy, the percentage prostate volume that received 100% of the prescribed minimal peripheral dose (V100) was 98.5%, and the rectal volume (mL) that received 100% of the prescribed dose (R100) was 0.02 mL. One year after seed implantation, the PSA level reached the nadir at 0.38 ng/mL. The PSA level then increased gradually. Forty-two months later, PSA recurrence was confirmed based on the Phoenix definition (nadir + 2 ng/mL). The patient was then observed without any salvage therapy because the dosimetric quality of the initial therapy was excellent and the possibility of PSA bounce was also considered. Fifty-eight months after seed implantation, the PSA level reached 5.11 ng/mL. A prostate saturation biopsy demonstrated no obvious recurrence inside the prostate. Systemic screening by lung and abdominal CT and bone scan did not detect distant metastases. On the other hand, T2-weighted and dynamic MRI images demonstrated a low intensity area at the base of the right seminal vesicle, which was very suspicious of recurrence (Figure A,B,C). We proposed salvage prostatectomy, salvage brachytherapy or androgen deprivation therapy to the patient. Sixty months after the initial therapy, he underwent salvage brachytherapy after providing informed consent. The prescribed dose of the salvage therapy was 145 Gy as same as the initial therapy. We used a radioactive source of 0.33 mC1 (13.1 MBq) for salvage brachytherapy (as same as initial brachytherapy). Prior to salvage brachytherapy, seminal vesicle biopsy was performed and the histological findings showed an adenocarcinoma with a Gleason score of 4 + 3 at the base of the right seminal vesicle. The base of right seminal vesicle was set as the planning target volume (PTV) with 1 cm margin. Seven needles were inserted into the PTV, and 2 or 3 sources were implanted through each needle. Finally 20 sources were implanted by the real-time planning method (Figure ). Regarding dosimetric parameters using CT images 1 month after salvage brachytherapy, seminal vesicle D90 was 280 Gy, seminal vesicle V100 was 100%, prostate D90 was 206.4 Gy, prostate V100 was 99.2%, the R100 was 0.26 mL, and the minimal dose (Gy) received by 30% of the urethra (UD30) was 285.7 Gy. The treatment time was 69 minutes. Since the PSA level rapidly decreased to 0.898 ng/mL by 1 month after seed implantation, he received no adjuvant therapy. Ten months after seed implantation, the PSA level reached 0.078 ng/mL. No adverse events were seen during the follow-up period.


A 43 years old female patient was previously submitted to laparoscopic gastric bypass for morbid obesity without co-morbidities. Seven months after the bariatric surgery the patient showed intense episodic epigastric abdominal pain that was aggravated by food ingestion and led to multiple visits to the emergency room.
One month after the onset of pain complaints, the patient was offered hospital admission for further evaluation. Physical examination was unremarkable and routine blood assessment of liver and pancreatic functions were normal. The upper endoscopy and esophageal-gastro-jejunal transit were normal; the abdominal CT and MRI were considered normal despite the presence of a mass in the excluded stomach, as it was ascribed to the anatomical rearrangement after the bypass surgery (Figure A). During hospital stay the patient did present any evidence of abdominal pain, complaints or need for analgesia; after formal psychiatric evaluation, a major depression was diagnosed and the patient was started on anti-depressants.
Five years after gastric bypass, due to ongoing epigastric pain complaints, abdominal CT and MRI were repeated, with subsequent diagnosis of a 4.5 cm of greater diameter subserosal neoplasm in the antrum (Figure B).
The patient underwent laparoscopic gastrectomy of the excluded stomach for suspected gastrointestinal stromal tumor (GIST) (Figure A). Gross examination of the specimen revealed a subserosal polypoid mass in the gastric antrum, which corresponded to a 4.5 cm cystic cavity of greater diameter with creamy yellowish thick content, growing in dependency of the gastric muscular layer (Figure B). The histology of the mass showed a flap of gastric wall with antral mucosa and a heterotopic pancreatic cist, while in the adipose tissue of the root of the greater omentum six other yellow and lobulated nodules were identified and dissected. All fragments corresponded histologicaly to pancreatic tissue with normal exocrine and endocrine distribution, as displayed by the immunohistochemistry staining for chromogranin A, insulin and glucagon expressing cells, as well as a low proliferation index as revealed by the Ki-67 staining, which are characteristic of the normal pancreatic tissue (Figure , A-F). After gastrectomy, the patient became asymptomatic and so has remained ever since.


A 58-year-old female was diagnosed with non-Hodgkin lymphoma in 1996 and was treated successfully with chemotherapy. She developed end-stage nonischemic cardiomyopathy from Adriamycin toxicity and underwent orthotic heart transplantation during June 2013. Three months into her transplantation, she developed shortness of breath and was found to have nonspecific pulmonary infiltrates and without any evidence of a pulmonary nodule on a CT of the chest. She was suspected to have invasive pulmonary aspergillosis (IAP) and was placed on oral voriconazole (400 mg bid). A flexible bronchoscopy (FB) was performed in a usual fashion under conscious sedation. Bronchoalveolar lavage (BAL) and five TBBx specimens were obtained from the right lower lobe (RLL) anterior segment under fluoroscopic guidance. There were no complications and the amount of bleeding was minimal. The histological examination of the biopsy revealed alveolated lung parenchyma with no pathologic diagnosis. The special stains for fungal organisms were negative.
A repeat chest X-ray on day 5 of the FB revealed a subtle opacity overlying the RLL in close proximity to oblique fissure. On day 13 of the procedure, a 9 mm RLL pulmonary nodule was detected on a repeat chest film. CT scan of the chest a week later showed a focal oval-cylindrical opacity at the peripheral aspect of RLL measuring 19 × 8 mm. Interestingly, the location of the opacity matched the site of TBBx. Besides, it showed resolution of the nonspecific pulmonary infiltrates (). The patient continued to show subjective improvement on empiric voriconazole therapy. In view of patient's stable pulmonary status, this nodule was thought to be related to the TBBx and no further diagnostic work-up was carried out. A repeat CT scan a month later, 7 weeks following the procedure, was unremarkable, except for the RLL PPN, which reduced to 16 × 6 mm in size. A follow-up CT scan of the chest at 3 months from the FB revealed total resolution of the pulmonary nodule () [].


A 46-year-old man (weight, 78 kg; height, 172 cm), who resided in the countryside of Parnaíba, Piauí, Brazil, consulted us for complaints of dizziness and frequent headaches. He was diagnosed with FH according to the clinical and laboratory criteria established in the Brazilian Guideline for Familial Hypercholesterolemia (based on the criteria of the Dutch Lipid Clinic Network) []. In addition, he had xanthomas on the elbows, soles of the feet, and Achilles tendons () and a family history of CAD, since his father died from acute myocardial infarction (the patient was unable to inform the occurrence of CAD in other family members). The patient had no other cardiovascular risk factors such as smoking, high blood pressure, and diabetes mellitus and was physically active. The patient's LDL-C and HDL-C levels were 545 and 53 mg/dL, respectively. His fasting glucose and triglyceride levels (85 and 158 mg/dL, resp.) were within normal limits.
The patient reported no use of lipid-lowering medication until the time of diagnosis. For socioeconomic reasons and/or lack of instruction, he had not sought medical care until when his symptoms eventually made him unable to work in the field. However, no symptoms of CAD were revealed.
Neck auscultation revealed a systolic murmur 3+/6+ in the neck, radiating to the skull. Electrocardiography, chest radiography, stress testing, Holter monitoring, and echocardiography revealed no abnormalities. Visual examination of the aortic root and aortic valve revealed no signs of their involvement (). On ultrasonography of the carotid arteries, we observed severe stenosis in the left internal carotid artery (LICA), with stenosis estimated between 70% and 90% (according to the criteria established by the European Carotid Surgery Trial and North American Symptomatic Carotid Endarterectomy Trial, as well as Dopplervelocimetric data) and moderate stenosis in the right internal carotid artery (RICA) estimated between 40% and 50% (). For the LICA, the peak-systolic (PSV) and end-diastolic velocity (EDV) cutoff values were 208.5 cm/s and 54.5 cm/s, respectively (Dex: 0.83 cm; Dint: 0.20 cm; %Std: 76.13); RICA PSV was 91.72 cm/s and RICA EDV was 37.37 cm/s (Dex: 0.74 cm; Dint: 0.44 cm; %Std: 41.27). We observed the presence of plaques in the anterior and posterior walls of the internal carotid artery and common carotid artery, which were characterized as bulky plates extending to the middle third of the internal coronary arteries (ICAs) and as predominantly echogenic and hyperechoic, with less than 50% of the area being echolucent with uneven surfaces, according to the Classification of Atherosclerotic Carotid Boards [].
A low-cholesterol diet was immediately recommended to the patient, and clinical treatment with statins was initiated. Concurrently, the patient underwent percutaneous revascularization by stent placement in the common and left internal carotid artery, and postoperative control showed adequate blood flow (). No intraoperative or postoperative complications were encountered. The patient remained asymptomatic, but the LDL-C levels remained high (320 mg/dL).


A 57-year-old woman presented with a 5-year history of a progressive foreign-body sensation in the throat and a change in her voice. She reported no dysphagia and/or dyspnea and she was a nonsmoker. Flexible laryngeal endoscopy () revealed a single, round tumor covered with smooth mucosa in the supraglottis. Although the tumor precluded visualization of the glottic gap, deliberate endoscopic manipulations revealed that the vocal cords were intact. To diagnose the epiglottic tumor in the simplest manner, we used forceps to partially puncture its anterior wall under local anesthesia () and noted that liquid contents did not drain from it. Therefore, it was diagnosed as solid rather than cystic. Subsequently, contrast-enhanced computed tomography (CT) scans () showed an approximately 4 cm diameter tumor with a wide base on the lingual surface of the epiglottis. The internal density of the tumor was consistent with adipose tissue and the CT contrast effects were only observed in the peritumor regions. Consequently, the tumor was suspected to be a lipoma that had developed on the lingual surface of the epiglottis. Considering its potential to obstruct the supraglottic airway, the giant tumor was immediately treated by surgical intervention.
We performed a tracheotomy on the conscious patient prior to inducing general anesthesia. Subsequently, we proceeded with microscopic laryngeal surgery using a cylindrical direct laryngoscope. However, the operating field was poorly visualized because the tumor was too large. Moreover, the instruments for microscopic laryngeal surgery were too small to manipulate the giant tumor. Because of the difficulty in completing the microscopic surgery, we proceeded with macroscopic surgery (). Following extirpation of the tumor, we inserted a direct laryngoscope to microscopically confirm that the surgical wounds were localized on the lingual surface of epiglottis, the epiglottic cartilage was not deformed, and the mucosal cutoff margins of the tumor were scarcely bleeding. Finally, we sharply trimmed the mucosal remnants of the tumor to cover the epiglottic wounds without necessitating sutures or fibrin glue. We maintained the tracheostomy tube after the surgery was completed and it was removed 3 days postoperatively because there was no swelling associated with the epiglottic surgical wounds. The patient was discharged 10 days postoperatively.
The surgical specimen was a clearly marginated yellowish tumor measuring 40 × 39 × 36 mm. Histopathologically, the tumor was composed of mature adipocytes covered with multiple fibrous capsules with no evidence of malignancy. These findings established a diagnosis of epiglottic lipoma on the lingual surface.
The 1-year postoperative course has been uneventful, and, subjectively, the symptoms associated with the throat have disappeared. There has been no recurrence of the epiglottic lipoma ().