Our patient was a 55-year-old female, who manifested shortness of breath, decreased effort tolerance, cough, and weight loss that began a few months earlier. She did not experience any fever, night sweats or other constitutional symptoms. She had no significant past medical history and never smoked or abused alcohol. On physical examination, she had stable vital signs and had no cyanosis, edema or lymphadenopathy. She had an increased air entry, dullness to percussion, and a reduced tactile vocal fremitus on the right side. The remainder of the examination was normal. She also had a normal blood count, basic metabolic panel, liver function tests, and coagulation profile. Her initial chest X-ray showed a large opacity in the right hemithorax. A subsequent chest CT demonstrated a 12 × 9 × 8 cm mass in the middle lobe of the right lung (fig. ). Based on the above findings, malignancy was suspected and bronchoscopy was performed, which showed a blunting at the carina, mucosal tram lines in both bronchi, and an external compression of the right lung, but endobronchial lesions were not observed. The histopathological examination of CT-guided transthoracic biopsy material was consistent with the thyroid transcription factor-1 (TFF-1) positive lung adenocarcinoma, and no epidermal growth factor receptor-activating mutations or echinoderm microtubule-associated protein like 4-anaplastic lymphoma kinase translocations were detected. Then, the patient was transfered to our department.
Distal organ metastases were not detected with whole-body bone scintigraphy and cranial CT; in the abdomen, a 5.3 cm mass was identified (fig. ). An exploratory laparotomy was performed which demonstrated several nodules in the greater omentum and the peritoneum (the largest lesion was 0.5 cm in diameter). A biopsy of the omental tumor revealed adenocarcinoma. The immunophenotype demonstrated positive TTF-1 and cytokeratin (CK) 7 staining (fig. ) and was negative for CK20. Those histologic findings corresponded with the primary lung cancer diagnosis and yielded an additional diagnosis of peritoneal metastases.
The patient was administered a chemotherapy based on carboplatin (AUC 6), paclitaxel (200 mg/m2) and bevacizumab (15 mg/kg) every 3 weeks. Following 6 courses of this treatement, a radiological evaluation was scheduled. A partial response was obtained and maintenance with bevacizumab was initiated. At 6 months after treatment, the patient presented brain metastases and received 30 Gy of palliative whole-brain radiotherapy. Therefore, a second line of chemotherapy based on pemetrexed (500 mg/m2) was administered every 3 weeks. Following 9 courses of chemotherapy, a clinical and radiological response was observed. Today, the patient is no longer undergoing chemotherapy and has not shown any symptoms of progression while waiting for a new evaluation.


A 44-year-old woman was admitted for chronic cough syndrome and refractory asthma criteria. The patient had a history of hypertension and morbid obesity with body mass index of 42 despite gastric reduction surgery 11 months earlier by means of sleeve gastrectomy. Since surgery, she had reported chronic cough with 10 recurrent exacerbations of dyspnea and wheezing requiring visits to the emergency services. In one of the visits, pneumonic infiltrates were observed in lung bases; after 12 days, they reappeared at another site and were therefore defined as migratory (see Figs. , ). Blood tests revealed a total IgE of 5.54 kU/L (n = 150 kU/L), and blood eosinophils of 225 (n < 400/mm3). Given the repeated asphyxial episodes, and despite inhaled corticosteroids and high-dose long-acting beta-adrenoceptor agonists, she was readmitted for further study. During her stay, she presented several asthma attacks with declines in peak flow exceeding 20%, and two sudden decreases of 40% and 60%, respectively. During the periods between attacks, the spirometric pattern remained within normal limits. Brittle asthma was diagnosed [].
Using laryngeal fibroscopy, the reflux finding score was 8 points (n < 7). Given these results and the lack of improvement with the addition of proton pump inhibitors (40 mg/bid), impedance–pH monitoring was performed. This test detected the existence of a severe mixed acid–nonacid esophageal reflux, with 75% of the events reaching the upper third of the esophagus and a significant increase in nonacid or weakly acid GER episodes. For all refluxes, there was a symptomatic association probability of 100% (n < 95%) and a symptom index of 100% (n < 50%; see Table ).
Given the evidence of uncontrolled GER with a clear temporal association between the onset of extraesophageal symptoms and the gastric reduction surgery performed 11 months earlier, the digestive tract was reoperated by open gastric bypass with incisional surgical repair and conversion from sleeve to bypass. A few days after surgery, and at 2 and 6 months after surgery, the patient presented no respiratory symptoms.


A 49-year-old female patient was admitted complaining of persistent pain and numbness in both legs for 4 months and a recently developed gait disturbance. These symptoms had intensified recently, and she was unable to walk without assistance. No remarkable disease or trauma history was noted. A neurological examination revealed bilateral numbness of the lower extremities that was particularly right-side dominant with an unclear dermatome. Hip flexion and extension motor grade was grade IV bilaterally. The pathological Babinski and ankle clonus reflexes were positive. Bladder and anal sphincter functions were preserved and motor and sensory functions of the upper extremities were normal. A laboratory examination and plain radiography of the cervical and thoracic spine were unremarkable. Magnetic resonance image (MRI) demonstrated a 1.7×1.4-cm lobulated mass in the C7-T1 dorsal space, probably the intra-dural extramedullary space (). T2-weighted MRI showed a low-signal-intensity elliptical lesion, which was enhanced peripherally on a gadolinium enhanced T1-weighted image. The spinal cord was severely compressed and displaced anteriorly by the mass lesion.
A C7-T1 cervical laminectomy and vertical incision in the dura were performed. No dural attached or adherent point was observed around the mass lesion when the intra-dural space was explored. The mass was covered with a white membrane, which was dissected and opened, revealing a 1.6×1.4-cm yellowish round mass (). Debulking and careful resection was performed using a microsurgical technique. A complete resection was achieved.
Spindle-shaped cells in a collagen-rich matrix were observed microscopically. The spindle tumor cells showed moderate nuclear polymorphism and formed parallel fascicles. Immunohistochemical staining revealed vimentin-positive, epithelial membrane antigen-negative, and S-100-protein-negative tissues (). These findings were consistent with a fibrous-type meningioma.
The leg numbness and gait disturbance showed gradual improvement postoperatively. The 1 week postoperative MRI revealed no residual tumor and no complications (). The patient was discharged without any complications. At the 1-month follow-up, the gait disturbance had improved, and she was able to walk without assistance.


A 14-year-old boy sustained a direct impact to his left shoulder due to an ice-hockey body-check. He complained acute pain and loss of function of his left shoulder. Initially the shoulder was immobilized in a shoulder sling and the patient was introduced to our department for shoulder and elbow surgery 10 days after the injury. Clinically, he presented with a prominent left acromioclavicular (AC) joint with pain on horizontal and vertical manipulation of the lateral clavicle, as well as marked tenderness over the coracoid process. Functional evaluation of the AC joint revealed a moderate superoinferior (3-5 mm) but no anteroposterior instability. Active range of motion (ROM) was restricted in every direction [ab-/adduction (Abd/Add): 50-0-10°; flexion/extension (F/E): 60-0-30°; external/internal rotation (ER/IR): 40-0-85°], passive ROM was normal. There was no evidence for an injury of brachial plexus or vascular compromise. The patient’s demands were complete and fast recovery with return to full sports as soon as possible, since he was playing ice-hockey in a first league team.
Computed tomography (CT) showed an AC joint separation with elevation of the lateral clavicle of 5 mm () and a fracture at the epiphysis of the coracoid base with 3 mm dislocation medially (). The magnetic resonance imaging (MRI) revealed an AC joint sprain with intact AC capsule and ligaments as well as intact coracoclavicular ligaments (CCL) ().
We started non-operative treatment on first presentation with immobilization in a Camo®Shoulder brace (OPED, Valley/Oberlaindern, Germany). This device allows resting of the 90° flexed elbow on a pad, thereby partially unloading the shoulder girdle, with fixed abduction of 15° and internal rotation of 45° (). At clinical and radiological follow-up 2 weeks later, the patient presented with persisting tenderness over the coracoid process but less tenderness over the AC joint. Active ROM was still markedly restricted (Abd/Add: 60-0-10°; F/E: 60-0-30°; ER/IR: 40-0-80°). The radiographs ruled out a secondary dislocation of the coracoid process. Passive rotation of the shoulder within the dynamic brace was allowed between 30 and 60° IR from week 4 and between 20 to 80° IR from week 5 (). At 8 weeks follow-up the patient presented completely pain-free with full ROM () and without tenderness on palpation over the coracoid or the AC joint. Clinical tests for the AC joint, rotator cuff and long biceps tendon revealed to be normal. After week 8 the patient resumed ice-hockey training and regained sports activity on first league level after 12 weeks. After 5 months he presented with mild symptoms of subcoracoid impingement which was successfully treated with physiotherapy within 6 weeks. A MRI confirmed complete bony healing of the coracoid fracture without malalignement, a coracohumeral distance of 13mm and signs of a mild subcoracoid bursitis (,).


A 54-year-old male reported to our department with history of swelling over the right side of face since 5 years. The patient reported insidious onset of swelling with a gradual progress to the present size. He also reported mild, intermittent, and dull pain in the region of swelling.
Clinical examination confirmed a solitary, oval, well-defined swelling measuring approximately 7 × 5 cm in the preauricular region []. The surface over the swelling was smooth without any secondary changes. It extended superioinferiorly from the level of outer canthus of the eye to inferior border of mandible and anterioposteriorly extended from 2 cm in front of anterior border of ramus to the mastoid notch []. On palpation, the swelling was firm, non-tender, free from skin, and mobile over the underlying structures. Mild pulsations could be appreciated from the swelling. There was no weakness of facial muscles or enlargement of swelling during mastication.
Ultrasonography revealed a large lobulated solid mass of 6 × 4 cm in the right parotid region with multiple vessels with low velocity flow and without any areas of calcification or cystic changes. Findings were suggestive of a low-grade vascular tumor.[] Computed tomography scan with contrast medium revealed a large well-circumscribed, solitary isodense lesion measuring 6.36 × 4.39 cm []. It was observed to be extending anteriorly over the superficial lobe of the parotid gland, with anterior displacement of parotid gland. A diagnosis of mixed parotid tumor was made.[] FNAC revealed numerous vascular channels with occasional singly scattered and single cluster of plump spindle cells. Based on these findings, a lesion of vascular origin was suggested.
Classical superficial parotidectomy was performed under general anesthesia using a modified Appiani incision. Tumor mass was dissected anteriorly and excised along with the superficial lobe of parotid gland []. Facial nerve was located using the anterograde method and all the branches of facial nerve were preserved. Post-operative recovery was uneventful. Upon six months of follow up, facial nerve function was intact and no sign of reoccurrence was observed.
Histopathological sections revealed connective tissue with numerous vascular spaces of variable sizes ranging from small capillary-like vessels to partly lined vessels. Around the endothelial cells, few vascular spaces showed a rim of circumscribed smooth muscle cells. The supporting connective tissue stroma appeared immature and fibromyxoid with sparse cellularity. Cells were plump or stellate-shaped with mild inflammatory cell response and composed predominantly of plasma cells and a few lymphoid follicles []. Immunohistochemistry revealed strong uptake of CD34 in the endothelial cells [].[] Findings were suggestive of a vascular lesion compatible with diagnosis of ENA.


We present the case of a 74-year-old Caucasian male presenting with a 2-year history of left knee pain. He described difficulty kneeling and descending stairs. On examination, a tender and mildly diffuse swelling was present over the left patella associated with mild erythema. The patient was able to perform a straight leg raise test and had full range of movement of the left knee. Ligamentous and meniscal testing was unremarkable as was examination of the ipsilateral hip.
Nineteen months earlier the patient had consulted his general practitioner complaining of left knee pain. Radiographs performed at that time were reported to show joint space narrowing within the patello-femoral compartment as well as early joint space narrowing within the medial compartment. A subchondral lucency was also noted within the patella which was described as a subarticular cyst secondary to the previously mentioned patello-femoral degeneration (Figs –).
The patient had a past history of prostate cancer, diagnosed in October 2005. He had been successfully treated and discharged from follow-up. In August 2007 he underwent excision of a basal cell carcinoma and had a permanent pacemaker for bradyarrhythmia. His only regular medication was a statin. Overall, he enjoyed good health and considered himself very active. There were no symptoms or signs of systemic disease.
Repeat radiographs performed by his general practitioner 19 months after initial presentation (Figs –) revealed a large lucency within the patella and a discontinuity in the anterior cortex of the inferior pole suggesting pathological fracture. A bone scan was arranged and demonstrated isolated abnormal tracer activity around the left patella (Fig. ). Due to the pacemaker a computed tomography (CT) scan was performed to further characterize the lesion. This confirmed a large lucent area occupying most of the patella with multiple areas of cortical disruption along its anterior border (Fig. ). Since the exact nature of the lesion could not be determined an ultrasound-guided biopsy was suggested but ultrasound screening revealed the lesion to be highly vascular (Fig. ). In view of this the radiologist performed a renal ultrasound, which revealed a large mass arising from the superior pole of the left kidney. Subsequent CT of the chest, abdomen and pelvis followed demonstrating the extent of disease. There was a large (13 × 8.5 × 9 cm) mass arising from the superior pole of the left kidney (Fig. ), the appearances of which were consistent with a primary RCC. Metastases were found in both adrenals and lungs, but no other bone metastases. Since diagnosis, the patient has had a left nephrectomy and is currently receiving radiotherapy and zolendronic acid treatment for the patella metastasis. At the time of writing this report the patient's patella lesion continues to be managed non-operatively.


A 49-year-old male patient who was suffering from aggravation of posterior neck pain, numbness in the left hand, pain in the left lower limb, and gait disturbance since 3 years visited our out-patient department. He had been diagnosed as having non-dysraphic cervical intradural lipoma and was operated in August 1990. This case was previously reported.
We had reviewed the patient's previous chart since he re-visited our out-patient department. According to the previous chart, he had been suffering from posterior neck pain for one year, and at that time, there were no neurologic symptoms except for neck pain. Physical examination did not reveal any stigmata such as dimples, hair, fistula, or any masses on his vertebral column. Previous preoperative cervical plain film images showed severe cervical lordosis and increased anteroposterior diameter of the spinal canal (). Previous computed tomography (CT) and magnetic resonance imaging (MRI) images showed a huge lipomatous mass, which was located intradurally and it encircled and compressed the spinal cord from the C2 to C7 level (, ). Although he was neurologically intact, we performed adequate debulking of the lipomatous mass with subtotal laminectomy from the C2 to C7 level in order to prevent neurological deterioration. Histopathological examination showed mature and uniform adipose cells without any cellular pleomorphism, suggesting the diagnosis of a benign lipoma.
When the patient re-visited our outpatient department, neurological examination showed slightly decreased motor power in the left lower limb (MRC grade 4/5), numbness in the left hand along the C6 and C7 dermatomes, and posterior neck pain radiating to the left shoulder. On radiologic examination, we found regrowth of the lipomatous mass from the C2 to C7 level, which encircled and compressed the cervical spinal cord. Previous subtotal laminectomy from C2 to C7 and severe cervical lordosis were also observed (, ).
We performed subtotal laminectomy of C1 in order to identify the normal elements of cervical dura because there were severe adhesions between paraspinal muscles, soft tissues and cervical dura. After that, we could perform dissection of the previous adhesions and could identify the cervical dura (). When the dura was opened, we found a huge, yellowish mass in the intradural space, which encircled and compressed the cervical spinal cord (). We performed subtotal debulking of the lipomatous mass using ultrasonic tissue ablation system paying careful attention to signals of intraoperative neurophysiological monitoring (IONM). There were no complications during IONM in the intraoperative course. We performed an adequate decompression of the spinal canal, and therefore we could suture the dura watertightly without duraplasty (). On the postoperative day 2, we found a cystic mass at the surgical site and it had gradually increased in size. On MRI, the cystic mass showed low signal intensity on T1-weighted images and high signal intensity on T2-weighted images; it suggested the presence of cerebrospinal fluid (CSF) (). We gently aspirated the cystic mass with a 24 gauge needle attached to the syringe, and then the surgical site was continuously compressed. After that, the cystic mass spontaneously disappeared. Histopathologic examination showed mature adipose tissue and fibrous tissue without cellular pleomorphism, suggesting the diagnosis of a benign lipoma ().
We are following up the patient for 24 months at the out-patient department, and his neurological symptoms have improved.


A 74-year-old man with a history of cerebral infarction was admitted with watery diarrhea that had lasted for 1 month. PEG had been performed 9 months prior to this admission. The patient's condition had been stable for 8 months after PEG. Vital signs and laboratory results upon admission were as follows: blood pressure, 80/40 mmHg; body temperature, 36.8℃; white blood cell count, 18,300/µL; hemoglobin level, 13.2 g/dL; BUN level, 10.3 mg/dL; creatinine level, 0.9 mg/dL; urine white blood cell count, high. His septic condition improved after adequate hydration and the administration of broad-spectrum antibiotics. However, the postprandial watery diarrhea persisted. On the 12th day of hospitalization, an upper endoscopy revealed a gastrocolic fistula and the internal bumper of the PEG tube was not seen (). The feeding tube was withdrawn after the upper endoscopy. On the same day, a colonoscopy also revealed a gastrocolic fistula in the transverse colon (). Several metal clips were applied to seal the gastrocolic fistula opening (). Abdominal CT performed before upper endoscopy showed that the bumper of the feeding tube had migrated into the colonic lumen () and revealed a fistula tract between the stomach and colon (). In a radiologic study using gastrografin administered through the nasogastric tube 1 week later, there was no leakage of dye (). The patient was discharged with a nasogastric tube for feeding after the external opening of the gastrocolocutaneous fistula was closed. The patient has been alive in a nursing home for 3 months after discharge from the hospital.


A 77-year-old man with a history of alcoholic dementia and intracerebral hemorrhage was referred by a nursing home to undergo an exchange of feeding tubes because his tube had suddenly loosened 3 days previously. He had undergone a PEG procedure 3 times and a simple exchange of PEG tubes twice. The first PEG procedure was performed 33 months earlier for enteral feeding. The second PEG procedure was performed 2 weeks after the first because the patient had forcibly pulled out the initial feeding tube. The second PEG procedure had to be newly done because the tract of the first PEG site was obstructed shortly. The site of the second PEG was close by the first PEG site. The patient's condition was stable for 27 months after the second PEG procedure was done. The third PEG procedure was performed 6 months prior to this consultation owing to buried-bumper syndrome. At that time, the patient presented with a loosened tube and abdominal pain. Because an upper endoscopy revealed that the feeding tube bumper was buried in the gastric wall (), the feeding tube was removed. The previous fistula tract required 4 days to close completely. Subsequently, a new gastrostomy was performed near the previous PEG site before discharge. The patient's condition had been stable for 6 months after the third PEG. Upon admission to our hospital, the patient's blood pressure was 90/60 mmHg and body temperature was 36.1℃. The blood test results were as follows: hemoglobin level, 3.7 g/dL; BUN level, 108.2 mg/dL; creatinine level, 5.5 mg/dL; estimated glomerular filtration rate, 10.7 mL/min/1.73 m2; and CRP, 229.99 mg/dL. An upper endoscopy revealed that the bumper of the feeding tube was deeply buried within the gastric wall, forming a hole (). A large space was observed when the scope was advanced into the hole, and brownish material was attached to the bumper (). These findings suggested gastric wall perforation resulting from a deeply buried bumper. The feeding tube was exchanged to seal the gastric wall opening, and the bumper of the tube was positioned in the gastric cavity. The patient was admitted to the intensive care unit. Because peritonitis and aspiration pneumonia caused a septic condition with acute kidney injury, medical therapies including hydration, blood transfusion, continuous renal replacement therapy, and broad-spectrum antibiotics were applied. In consideration of the patient's condition, medical treatment, including total parenteral nutrition, was continued rather than using a surgical option. The feeding tube was withdrawn when pus discharged from the gastrostomy site on the 10th day of hospitalization. The patient's condition improved for a while, and the external opening of the colocutaneous fistula was nearly closed 3 weeks after admission. According to his family's request to insert a feeding tube through the gastrostomy tract, an upper endoscopy was performed again. The gastric wall opening was still unhealed. When the scope was advanced into the opening, the lumen of the transverse colon and a bluish liver shadow were identified (). The diagnosis of a gastrocolocutaneous fistula was confirmed at that time. The patient died of multiple organ failure caused by pneumonia aggravation 1 week later.


A 59-year-old woman presented with severe neck pain and stiffness of a few days duration as well as moderate discomfort when swallowing. She had no trauma and no current or recent febrile upper respiratory tract infections. On physical examination, swelling and tenderness of the sternocleidomastoid was found bilaterally. Neck pain was induced by extension and reduced by flexion. Range of motion (ROM) of the cervical spine was extremely limited. Visual inspection of the pharynx and ultrasound examination of her neck revealed no pharyngitis, goiter, or lymphadenitis. Her blood pressure, pulse, respiratory rate, and temperature were within normal limits. White blood cell count was 6,160 cells/mm3 and C-reactive protein level was 1.2 mg/dL. Lateral radiographs of the cervical spine revealed a large calcium deposit anterior to the C1-C2 joint and swelling of the prevertebral soft tissue from C1 to C5 (). Neck CT scans revealed a low density area with the appearance of a convex lens in the retropharyngeal gap, but the contours of the longuscolli and longuscapitis muscles were clear (). MRI showed fluid in the retropharyngeal gap between C1 and C6 (). There were no findings of spondylitis, epidural abscess, or tumor.
A diagnosis of acute calcific tendinitis of the longus-colli was made, and a soft collar and non-steroidal anti-inflammatory drug (NSAID) were prescribed, without antibiotics. Two weeks later, neck pain, dysphagia, and ROM limitation were all reduced, and plain X-ray films taken at 3 weeks after presentation revealed reduced retropharyngeal soft-tissue swelling and almost complete disappearance of the amorphous calcification anterior to C1-C2 (). At 4 months after presentation, the calcium deposit and all of the abovementioned symptoms had resolved completely (). At 6months, fluid in the retropharyngeal gap between C1 and C6 was no longer evident on MRI ().


Case 16. A 24-year-old male patient transferred from the local hospital and complained of severe back and leg pain (). He had a history of previous L5-S1 discectomy and magnetic resonance images (MRI) suggest L5-S1 discitis. The ESR and CRP at the visit of our institute were 20mm/hr and 44.1mg/L, respectively. He complained of aggravating back and leg pain despite antibiotic treatment without bacterial identification for 6 weeks in the local clinic and the CRP value was not controlled. On the follow-up MRI and CT scan, spondylitisprogressed to L5 and S1 vertebral body and bony destruction was exacerbated. We thought infection is not controlled and persistent, that is, treatment failure, surgical treatment was performed. The anterior radical debridement of all infected and necrotic tissues including the infected vertebral body and soft tissues and anterior column reconstruction with freeze-dried structural allograft and staged posterior instrumentation were performed. Intraoperatively, upper portion of L5 vertebral body showed granulated change and no visible the healthy bleeding bone, therefore the corpectomy was extended to L5 totally to allow for subsequent bone fusion. Postoperative follow-up lumbar plain X-ray and CT scan taken at 12 months did not show graft failure. But, bone fusion was not yet achieved.


Case 21. A 69-year-old female patient complained of severe back pain. The patient had a history of previous spinal nerve root block in the lumbar spine (). The ESR and CRP at the time of hospital visit were 132mm/hr and 45.2mg/L, respectively. Initial MRI suggests spondylodiscitis of L3 and L4. Antibiotics were administered depending on the results of tissue culture for which needle aspiration. Back pain was quite improved and the CRP value was showed a decrease trend to begin with, but she complained of more deteriorating back pain and left lower extremity radiculopathic pain which had never been seen before, and the CRP value was elevated to 54.5mg/L at the time of 4 weeks after the antibiotics administration. On the follow-up MRI and CT scan, severe destruction of end plate and vertebral body at L3 and 4 and paravertebral and epidural inflammation worsened than initial images. The anterior radical debridement of infected and necrotic tissues including the infected vertebral body and soft tissues and vertebral column reconstruction with freeze-dried structural allograft and staged posterior instrumentation were performed. Intraoperatively, residual upper portion of L3 vertebral body and lower portion of L4 vertebral body after corpectomy showed yellowish-granulated change and no visible the healthy bleeding bone, therefore the corpectomy was extended to L3 and 4 totally to allow for good fusion bed. Postoperative follow-up lumbar plain X-ray and CT scan taken at 12 months show no graft problem and an ongoing bone fusion between the structural allograft and the vertebral body interface. Bone fusion between the cancellous allograft filling in the structural allograft and the vertebral body was also observed on the CT scan.


In 2001, a 58-year-old patient was primarily treated with radical prostatectomy for a pT3b (seminal vesicle invasion), pN0, M0 Gleason score of 9 (4 + 5) prostate adenocarcinoma. Because of positive radical margins, adjuvant, postoperative radiotherapy on the prostate bed was performed. Intermittent androgen deprivation with a luteinizing hormone-releasing hormone analogue was started in 2003 for rising serum prostate specific antigen (PSA) levels. In 2009, bicalutamide was added because of a castrate-resistant disease without any detectable metastases. Asymptomatic bone metastases occurred in 2012. Zoledronic acid was administered and the patient was thereafter included in a clinical trial assessing the efficacy of a monoclonal antibody targeting the αv subunit of human integrins. Seventeen months later, i.e. in September 2013, the disease evolved with new bone lesions and the appearance of retroperitoneal and mediastinal lymph nodes along with lung metastases. At the follow-up consultation, the patient presented with spontaneous cutaneous petechiae. PSA increased to 485 ng/ml. Platelet count was 29,000/ml (normal range: 150,000–300,000/ml), hemoglobin 10.8 g/dl (normal range: 12–14 g/dl), fibrinogen 0.9 g/l (normal range: 2–4 g/l), prothrombin time 69%, and a positive D-dimer test at 7,909 ng/ml (normal value: <200). The patient started AA (1 g/day in combination with prednisone 10 mg/day). On day 7, platelet count increased to 107,000/ml, fibrinogen rose to 2.8 g/l and coagulation blood tests normalized while PSA decreased to 190 ng/ml. Nadir PSA reached 6.4 ng/ml after 4 months of AA. At the 8-month follow-up and after AA treatment, the patient had a good performance status with normal hematological parameters, but his PSA had risen to 11 ng/ml.


The patient was a 20-year-old male with a 10-month history of pain in the cervical, sacroiliac, and peripheral joints accompanied by a skin rash. He had generalized weakness, anorexia, and intermittent, low-grade fever. The pain in the joints began in the sacroiliac and then extended to the peripheral and upper cervical spine. Also, he had pain in the upper cervical spine, but other parts of his spine were asymptomatic. He had morning stiffness for more than three hours, and his symptoms were worse at night and when he was resting. All of his symptoms progressed during the 10-month period, and he was unable to work. There was no sphincter disturbance.
The patient's head was tilted mildly to right and flexed. The range of motion of the peripheral joints was decreased, and movement was accompanied by tenderness and pain in most of the joints. The muscle strength of the upper extremities was unaffected, and there was no neurological deficit; however, the lower extremities had mild paraparesis (muscle strength score of 4, based on a five-point scale, in both distal and proximal of the lower limbs) with severe pain associated with all movements (passive and active). There was no hyperreflexia or signs of upper motor neuron issues. Scaling erythematous skin lesions was detected on the extensor surfaces of the extremities (keratoderma blennorrhagica). The patient indicated that he had short-term, bilateral red eye and dysuria before his admission to the hospital. Sausage-form digits (dactylitis) were observed on both hands. He had nail involvement in the form of nail hyperkeratosis and onycholysis. Also, he had balanitis circinata (also known as circinate balanitis) that was manifested as annular dermatitis of the glans penis.
The patient had a history of unsafe sexual contact with subsequent bloody and mucopurulent discharge from the penis and other symptoms of sexually-transmitted disease. The familial history of this patient was negative.
He underwent skin lesion biopsy that revealed psoriaform tissue reaction with intra-epidermal pustule formation, associated with keratoderma blennorrhagicum (Fig. ). There were some significant abnormal findings in his laboratory data, such as leukocytosis, thrombocytosis, elevated erythrocyte sedimentation rate (110) and C-reactive protein [], urine pyorrhea, and positive HLA-B27. He had negative rheumatoid factor (RF) testing.
The pelvic X-ray and MRI indicated the presence of bilateral sacroiliitis (Fig. , ). The cervical spine X-ray showed that there was a 27 mm anterior displacement of the cranio-atlantal complex on the axis (Fig. ). The MRI of the cervical spine with and without contrast indicated the presence of tearing of the transverse ligament accompanied by posterior displacement of the odontoid process (Fig. ) and cervical cord compression from the left and C1 rotation to the right (Figs. , ). The anterior unilateral displacement of C1 on C2 was greater than 5 mm. Thus, the patient's AARS was grade 3 based on Fielding grading [] and unilateral anterior based on the White and Panjabi classification []. Dynamic imaging showed that there was significant angulation in the atlanto-axial junction (Fig. , ).
The patient underwent five pounds of traction with Gardner-Wells tongs in a neutral position. Because the dislocation of the upper cervical spine was reducible with traction (Fig. ), we decided to perform open reduction surgery from the posterior, based on the diagnosis of AARS. Thus, before the patient was anesthetized, he was intubated and placed in a prone position with five pounds traction; the forces on all four limbs were rechecked, and he underwent generalized anesthesia. The intraoperative reduction was performed using a fluoroscopic guide. Then, C1 bilateral laminar hooks and C2 bilateral pedicular screws were placed and connected with rods; also, an autograft and allograft bone for fusion was added. Control imaging was obtained intraoperatively.
Immediately after the operation and at the three-month follow-up examination, the patient had no neurological deficits, no instability or canal compromise in imaging (Fig. ), and no cervical manifestation, except limited cervical rotation.


A 64-year-old man visited Jichi Medical University Hospital complaining of left eye visual loss and floaters in the right eye. He was referred to us by the Department of Allergy and Rheumatism of our hospital, having been administrated 40 mg of oral prednisolone for more than 6 weeks as treatment for interstitial pneumonia accompanying rheumatoid arthritis. His medical history included metastatic rectal cancer, intestinal obstruction, and pericarditis.
Best-corrected visual acuity (BCVA) was 20/20 in the right eye and 8/200 in the left eye. Funduscopy revealed round yellowish-white subretinal lesions with retinal hemorrhage in both eyes ().
Laboratory studies showed elevated titers of the anti-Toxoplasma, anti-Herpes simplex virus IgG, anti-Herpes simplex virus IgM, and anti-cytomegalovirus IgG antibodies, as well as low cell lymphocyte and CD4-positive lymphocyte counts. In contrast, the white blood cell count, C-reactive protein, and β-D glucan levels were not increased.
No abnormalities were noted on blood culture or whole-body contrast computed tomography.
Administration of oral anti-Toxoplasma (acetylspiramycin 1,800 mg/day) and anti-viral drugs (valganciclovir 1,800 mg/day) was initiated. However, the lesions expanded in both eyes.
Diagnostic vitrectomy of the left eye was performed 4 weeks after the initial examination. Although the vitreous fluid sample was subjected to broad-range PCR, multiplex PCR, and other culture tests, no positive results were obtained.
Based on the fundus findings, broad-spectrum antibiotic treatment (imipenem/cilastatin 2,000 mg/day and amikacin 600 mg/day) was initiated, under suspicion of bacterial infections including Nocardia. However, the lesions did not improve. Thus, antibiotics (ceftazidime 2 mg, vancomycin 1 mg, and voriconazole 0.05 mg/0.1 mL each) were injected into the vitreous cavity 6 weeks after the initial examination, and a vitreous tap was simultaneously performed. Broad-range PCR of the vitreous fluid showed 3.75×104 copies/mL of bacterial 16s ribosomal RNA, strongly suggesting bacterial infection. Albeit with antibiotic treatment, the lesions further deteriorated BCVA to light perception ().
Seven weeks after the initial examination, exenteration of the left eye was performed. Immediately after the operation, microscopic observation of the removed and then Gram-stained tissue revealed Gram-negative rods. However, no bacteria corresponding to these organisms grew in culture of either the removed tissue or vitreous fluid, and only C. albicans was isolated. Sheep blood agar and chocolate agar were used for culture and intubated with 5% carbon dioxide at 35°C for 48 hours. The picture of the Gram-stained sample was taken from this culture (, left). Then, the colonies grown on these cultures were harvested and cultivated on the CHROMagar® for identification. The picture of the agar plate shows the colonies that are green in color on the CHROMagar® suggesting the species of C. albicans based on its color (, right).
Based on these findings, mixed bacterial and fungal infection was considered, and simultaneous systemic administrations of antibiotics (levofloxacin 500 mg/day) and maximum-dose antifungal agent (fosfluconazole 600 mg/day) were initiated. In addition, antibiotics (ceftazidime 2 mg, vancomycin 1 mg, and voriconazole 0.05 mg/0.1 mL each) were injected into the vitreous cavity of the right eye three times from 8 weeks to 10 weeks after the initial examination. Broad-range PCR of the vitreous fluid obtained from simultaneous vitreous tap 8 weeks after the initial examination revealed 7.19×104 copies/mL of bacterial 16s ribosomal RNA, and 1.85×105 copies/mL of fungal 28s ribosomal RNA, suggesting a fungus-dominant mixed infection.
The subretinal abscess in the right eye expanded, approaching the central fovea (), and BCVA decreased to 2/200. Vitrectomy combined with cataract surgery and silicone oil injection was performed 10 weeks after the initial examination. C. albicans was detected by vitreous fluid culture. Systemic administration of the antifungal agent was continued after surgery, and the lesions healed under silicone oil (). The fundus had stabilized 1 year and 4 months after the initial examination, and BCVA was maintained at 4/200.


A 26-year-old female patient reported to the Dental Department complaining about the unesthetic appearance of her face, since eight years []. History revealed that eight years back, the patient underwent surgery in the neck region for a cyst and one to two days after surgery the patient got a swelling on the face. The swelling was of the same size since then and there was no pain associated with the swelling. There was no history of difficulty in breathing or eating.
On examination, the face of the patient appeared asymmetrical, bilaterally, with a growth on the right side of the face. The skin over the right side of her face was thickened and hyperpigmented, with a soft and loose hanging overgrowth. There were numerous small and large, sessile and pedunculated nodular growths localized on the right side of her neck, with dark-colored underlying skin. The skin over the right cheek and chin area showed coffee-colored pigmentation (café-au-lait spots) [].
There was a soft, compressible, nontender growth on the lower lip. The right ear showed disfigurement and hypertrophy, with thickened cartilage and overlying hyperpigmented skin [].
Intraorally there were numerous soft tissue growths with a sessile base seen on the right buccal mucosa [] and tongue []. The growths were soft, compressible, and nontender.
Laboratory findings revealed a raised erythrocyte sedimentation rate (ESR) (70 mm in the first hour), a decreased hemoglobin level (7.5 gm%), and the enzyme-linked immunosorbent assay (ELISA) was found to be nonreactive.
A panoramic radiograph disclosed asymmetric enlargement of the right side of the mandible, with loss of cortication of the lower border in the midline area, expansion of the body of the mandible causing displacement of the teeth, seen on right side, expansion of the right inferior alveolar nerve canal, enlarged mental foramen, resorption of the roots of the mandibular right teeth, and loss of the right antegonial notch. A deficiency was seen on the right side of the maxilla, with crowding of the posterior teeth [].
The right lateral oblique radiograph revealed a deep sigmoid notch, loss of the antegonial notch, an enlarged and rounded coronoid process, with resorption of the mandibular right teeth [].
Subsequently, biopsy of one of the nodules was taken and sent for histopathological examination.
The histopathological slide revealed a non-encapsulated tumor of the dermis with a normal overlying epidermis. The tumor consisted of loosely spaced spindle cells and wavy collagenous strands in a clear matrix [].


The patient was a 39-year-old female who had a mass in the right parotid region. The mass was found 2 months previously and had subsequently showed a gradual increase in size. No history of trauma or injury was identified. On physical examination, a firm, mobile, well-circumscribed mass was palpable in the subcutaneous tissue of the right cheek. No nerve paralysis or paresthesia was identified. At the same time, the patient was diagnosed with papillary carcinoma in the right thyroid gland by fine needle aspiration cytology (FNAC). Before the patient underwent a right hemithyroidectomy, ultrasonography and FNAC were performed. Ultrasonography showed a 2.3-cm-sized irregular shaped mass in the right parotid gland with ill-defined margins. Subsequently, computed tomography scan showed a 1.9×1.3×1.2-cm-sized, heterogeneously enhancing lobulated mass (). Laboratory examination data were within normal limits. FNAC was performed for the mass of the right parotid region.
Papanicolaou staining from the aspirate showed moderately increased cellularity and was composed of isolated and cohesive cells with loose fibrous stroma. In cohesive parts, the cells consisted of short bundles or fascicles that did not form a characteristic pattern of arrangement. Most of the cells had oval or spindle-shaped nuclei and relatively abundant cytoplasm with rather indistinct cytoplasmic outline. Some cells had plasmacytoid features. Cellular pleomorphism was minimal. Inflammatory cells were also diffusely scattered (). Neither necrosis nor brisk mitotic activity was seen. Giemsa staining revealed that the mucoid-like stromal materials were metachromatic, which was reminiscent of the fibromyxoid matrix that is commonly seen in PA. Therefore, PA was initially diagnosed based on the cytologic findings.
After FNAC diagnosis of the mass, superficial parotidectomy was performed for treatment and histological confirmation. The removed specimen was 2.3 g in weight and 2.4×1.8×1.2 cm in dimensions. On section, the tumor was well demarcated, but not encapsulated. The cut surface of the mass was solid and gray white ().
Histologically, the mass was predominantly composed of relatively uniform spindle cells which showed moderate cellularity. These spindle-shaped cells were arranged in short fascicles arranged in haphazard pattern. Focally, a storiform growth pattern was also noted. The tumor cells had centrally located round to oval nuclei with finely granular chromatin and one or two inconspicuous nucleoli. The stromal part was predominantly collagenous with focal myxoid areas. Glands or squamous structures were not noted. Some inflammatory cells including lymphocytes, neutrophils, and histiocytes were found. Many small thin-walled vessels and extravasated erythrocytes were scattered diffusely. Regarding mitotic count, about 8 per 10 high power fields were found, but no atypical mitotic figure was identified (–). There was no necrosis. Combined with these histologic findings, NF was diagnosed. Additionally, immunohistochemical staining was performed. The spindle cells showed positivity to vimentin (1:1,000, V9, Zymed, South San Francisco, CA, USA), smooth muscle actin (1A4, 1:400, Dako, Glostrup, Denmark), but negativity to S-100 (1:1,000, polyclonal antibody, Novocastra, Newcastle Upon Tyne, UK), desmin (1:200, D33, Dako), CD34 (1:400, QBEnd-10, Dako), and pan-cytokeratin (1:500, AE1&AE3, Labvision, Fremont, CA, USA) (–). The results of immunohistochemical staining supported the diagnosis of NF


A 50-year-old-male, presented with the complaints of left flank pain of 1 month duration. He denied having any constitutional symptoms, voiding difficulty, ejaculatory disturbances, hematuria or constipation.
Digital rectal examination revealed a mass in the region of left seminal vesicle separate from the prostate. The total leukocyte counts were within the normal limits, serum prostate specific antigen was 0.759 ng/ml and urine culture was sterile. Serum urea and creatinine values were 20 mg/dl (8-23 mg/dL) was 1 mg/dl (0.6-1.2 mg/dL), respectively. Repeat transabdominal and transrectal ultrasonography (TRUS) [] revealed a well-defined mass lesion, measuring 13.5 cm × 9.9 cm × 5.4 cm in size, posterior to the urinary bladder with homogeneous, hypoechoic echotexture in the region of left seminal vesicle. Prostate was normal in size and echotexture. There was moderate left hydroureteronephrosis with near normal renal parenchymal thickness and echotexture. The distal ureter was disappearing into the mass lesion. Computed tomography (CT) with contrast [Figure and ] revealed a mass with mixed cystic and solid densities in the location of left seminal vesicle. The lesion showed good post-contrast enhancement and no evidence of calcification. The terminal part of the left ureter appeared embedded within the mass lesion. The lesion was seen separate from the prostate and the left seminal vesicle wasn’t distinctly visible on the TRUS and the CT scans. Radiological opinion in view of the well-defined nature, solid-cystic, homogeneous and hypoechoic texture was that of a benign mass lesion. Since TRUS and CT gave adequate anatomical information of the lesion, magnetic resonance imaging (MRI) was not requested for, though that would have been the next logical step. Cystoscopy showed a trigonal bulge in the left side. Retrograde catheterization was not possible implying obstruction and or distortion of the juxtavesical ureter. Per-rectal fine needle aspiration of the mass yielded only blood and cytology of the same was inconclusive.
The lesion was explored through a lower midline incision on the abdomen. A huge mass lesion was encountered in the retrovesical location, obscuring the left seminal vesicle and involving the left ureter causing proximal dilatation. In the beginning ureter was liberated by meticulous dissection. This was followed by decompression of the tumor by scooping out of myxoid, jelly-like material after a capsulotomy. The capsule was carefully excised throughout taking care to preserve the neurovascular structure at the bladder neck. No separate seminal vesicle was made out at the culmination of tumor excision, suggesting that the former structure is the organ of origin. The right seminal vesicle was normal in appearance.
There were no post-operative complications and patient was discharged on 10th post-operative day. Histopathological examination revealed a benign schwannoma [] with degenerative changes such as foamy macrophages [] and cholesterol clefts [] and characteristic Verocay bodies []. Multiple sections did not reveal any seminal vesicle tissue within the tumor, suggesting that the tumor had completely effaced the organ. Patient was voiding well without incontinence and his erectile and ejaculatory functions are preserved. There has been no recurrence of tumor until date, that is, 2 years and 10 months after surgery.


A 39-year-old female presented with two months history of urgency, dysuria, lower abdominal pain and lower abdominal swelling. There was no history of hematuria. On physical examination a hard hypogastric mass was present fixed to the rectus muscle. It was bimanually palpable. Routine blood investigations were normal. Urine routine was normal and culture was sterile. Ultrasonography showed a heterogeneous mass lesion from the anterior wall of the bladder. Contrast-enhanced computerized tomography (CECT) abdomen showed a heterogeneous enhancing mass 8.8 by 4.4 cm mass arising from the anterior bladder wall and dome, extending to the umbilicus with infiltration of the overlying parietal wall []. Cystoscopy revealed extensive growth involving the entire anterior wall and dome of the bladder extending to posterior wall. A biopsy done revealed cystitis with focal areas suggestive of urothelial neoplasia of unknown malignant potential. Metastatic workup was normal. As we could not rule out the presence of bladder tumor and due to the large size on the lesion, we proceeded with radical cystectomy with ileal conduit. The mass was found to be arising from the entire wall of the bladder and was infiltrating into the posterior rectus sheath and muscle, which needed to be partly resected along with the specimen []. Histopathology revealed cystitis cystica with xanthogranulomatous cystitis (XC) of the entire bladder wall [Figures and ]. There was no malignancy or urachal remnant. The post-operative course was uneventful and at 1 year follow up the patient is doing well.


A 66-year-old woman visited the emergency department with a sudden onset of severe headache. She had no underlying disease, no history of anticoagulant therapy, no hematological coagulopathy and no history of trauma. She only underwent a mastoidotomy due to chronic otitis media at our institute one week before admission. She was alert but had a severe, worsening headache of sudden onset and neck stiffness. No neurologic abnormalities were found in the cranial nerves and cerebellar system. According to her medical history and neurologic examination, we conducted a brain computed tomography (CT) under the impression of acute subarachnoid hemorrhage, but it was unremarkable. Next, we conducted spinal tapping and she showed positive on 3 tube test. We then performed an angiography CT and 4 vessel angiography. But, no definite intracranial aneurysms could be found. The tentative diagnosis of non-aneurysmal subarachnoid hemorrhage was made initially. We hospitalized the patient, continued the supportive care and conservative management. Until hospital day 5, the headache was improved but posterior neck pain and neck stiffness persisted. We performed a cervical spine magnetic resonance imaging (MRI) and found a large amount of subacute subdural hematoma on C7 to T4 level (). We continued the supportive care as the patient had no neurologic deficit but only pain and the symptoms were gradually improved. She was discharged on hospital day 15. A follow up MRI was performed after 4 months. The hematoma was nearly disappeared and the patient presented no residual symptoms ().


A 34-year-old male, a computer engineer by profession, was found drowsy at home and was referred to a nearby medical center. There was history of a recent business trip to Greece which took almost 6 weeks. He was suffering from headache and fever and was drowsy when admitted in the hospital [modified Rankin scale (MRS) =2]. Preliminary workups were all negative and brain computed tomography scan (CTS) showed a small left frontal opercular intracerebral hemorrhage (ICH) and intraventricular hemorrhage (IVH) accompanied with mild hydrocephalus []. He refused to stay in the hospital for further investigations after improvement of his mental state. Headache, fever, and intermittent changes in his state of consciousness recurred after a month and he was admitted in our center after another attack of severe headache. At the time of admission, he was stuporous with mild right hemiparesis and pure motor dysphasia (MRS = 4). Brain CTS showed a left frontal opercular ICH []. Four-vessel digital subtraction angiography (DSA) showed a left distal middle cerebral artery (MCA) aneurysm that was 12 × 9 mm in diameter and without an identifiable neck []. This aneurysm was visible in both anteroposterior (AP) and lateral views even though there were two other ill-defined aneurysm blebs only seen in the lateral view of the angiogram. These two blebs were located along the more proximal branches of M3 segment of left MCA [].
Considering the past history of fever, headache, detectable neck rigidity, and ICH, the first impression was rupture of a mycotic aneurysm. None of the aneurysms had well-identifiable neck in the angiogram. It was planned to treat the presumable mycotic lesions with a trial of wide-spectrum antibiotic therapy including Vancomycin 1 g twice/day + Rifampin 600 mg three times/day and Tazocin 4.5 g three times/day. All of the preclinical studies were negative except for the following: erythrocyte sedimentation rate (ESR) =28, C-reactive protein (CRP) =3 plus, and positive serology for brucellosis (direct Coomb's test/Brucella micro agglutination test) both in his plasma and CSF with the agglutinin titer of 1/160 and 1/80, respectively. CSF analysis was compatible with partially treated meningitis: WBC = 500/mm3, polymorphoneuclears 60%, lymphocytes = 40%, protein = 55.3mg/dl, and sugar = 56 mg/dl.
His general condition improved moderately after 3 weeks of antibiotic therapy (MRS 3), but intermittent fever persisted and the CSF picture did not change notably. A brain computed tomographic angiography (CTA) did not show any regression in the size of the distal MCA aneurysm. The other two ill-defined aneurysms noted in the previous DSA were well elucidated in the new CTA [].
Considering the available facilities, it was decided to treat the aneurysms surgically and it was planned for either clipping-only or excision with reconstruction bypass surgery of the aneurysms according to the findings during surgery. A left posterior temporoparietal craniotomy was done after 30 days of antibiotic therapy. The dura was opened, taking care of the arachnoidal adhesions visible beneath the dura, and attached to one of the cortically exposed aneurysms. A small aneurysm was located along one of the cortical branches of M4, along the superior temporal gyrus with adhesions in the surrounding subarachnoid space. The arachnoid could be dissected sharply. The aneurysm was a small one located on a distal M4 branch, without a clippable neck. It was coagulated and excised. The second aneurysm was located along M3 segment of the frontal branch of MCA. It was dissected, clipped, and resected. The clip could be removed after micro-coagulation of the parent artery [Figure and ]. The more distal dissection of Sylvian fissure revealed a third small aneurysm located more distally along the M4 segment of MCA, which was dissected, clipped, coagulated, and excised. The histopathological examination of the resected aneurysms was compatible with “mycotic aneurysm” [Figure –].
The patient's postoperative course was uneventful. Considering the positive serology for brucellosis both in plasma and CSF, the anti-brucella antibiotic regimen was continued. The patient could be discharged after 1 month with improvement of fever and stuttering (MRS 2). The anti-brucella medications were continued for another 6 weeks and negative serology tests achieved. The postoperative CTA performed after 1 year confirmed no aneurysmal dilatation along the course of MCA [].


A three-year-old boy presented with progressive loss of scalp hair, eyebrows, and eyelashes since six months of age along with stunted growth as a major complaint. The child's antenatal history was normal but was born to third-degree consanguineously married parents. On examination, he had a senile look with prominent eyes, sparse hair with patches of alopecia and visible veins over the scalp, beaked nose, and receded chin []. His voice was high pitched and growth was stunted as parameters were less than the third percentile. Teeth and genitals were normal and intelligence quotient (IQ) was corresponding to the age. Hands were short and clawed with thickening and hardening of the skin over the knuckles with racquet nails. The ribs were prominent and anterior fontanelle was open. There was mottled pigmentation and sclerodermatous changes over the trunk and lower limbs []. Lower limbs also had prominence of knees and slight valgus deformity [Figures and ].
Routine investigations were within normal limits. The serum lipid profile showed a decrease in high-density lipoproteins (HDL). X-ray of the chest showed a pyriform thorax, overcrowding of proximal ribs, and short clavicle with pointed lateral ends []. X-ray of the skull showed diastasis of the sutures and prognathism []. X-rays of hands and feet showed acro-osteolysis of phalanges and tarsals [].


A 51-year-old Chinese female presenting with general weakness and pancytopenia for half a year was admitted to our hospital. A complete blood cell count analysis showed mild anemia, with a hemoglobin level of 87 g/L, white blood cell (WBC) count of 2.35×10/L, and platelet count of 23×10/L. Myelodysplastic syndrome (refractory cytopenia with multilineage dysplasia) was diagnosed by bone marrow examination, and karyotyping revealed 20q-:29.2% (73/250). After being conditioned with fludarabine, busulfan, high-dose cyclophosphamide, and antithymocyte globulin as a preparative regimen, allogeneic peripheral blood stem cell transplantation from her human leukocyte antigen (HLA)-matched sibling was performed. Engraftment was apparent on day 16 after transplantation, with total leukocytes recovering, from less than 0.5×10/L before transplantation to over 1×10/L. Prophylaxis for GVHD consisted of cyclosporin A, Mycophen, and high-dose dexamethasone. Despite these intensive efforts, by day 30, the patient had developed watery diarrhea, burst oral mucosa, skin rash, and abnormal liver function. Her alanine aminotransferase (ALT) and aspartate aminotransferase (AST) of serum were 301 IU/L and 106 IU/L, respectively, and her serum albumin was only 28 g/L, which was lower than the normal value. These symptoms and laboratory indexes strongly suggested the onset of acute GVHD, so the patient was additionally given supporting treatment, including magnesium isoglycyrrhizinate, omeprazole, berberine, and Smecta®, to improve liver function, protect the stomach, and control the symptom of diarrhea.
Cytomegalovirus (CMV) infection was diagnosed at day 80 after transplantation, with the level more than 5×10 Copies/mL, so the patient was treated with ganciclovir. The patient started GI bleeding at day 90, accompanied with abdominal cramping and bloody diarrhea of total 400 to 500 mL approximately five times per day. Multiple transfusions of red blood cell (RBC) concentrates, platelet, fresh frozen plasma, high-dose prednisolone, broad-spectrum antibiotics, antiviral coverage, and even daily hemostatic agents were utilized. The patient received therapeutic doses of RBC concentrates and platelet transfusions for more than 20 consecutive days after the symptom of bloody diarrhea began. However, despite all these efforts, the bloody diarrhea could not be stopped.
Upper and lower endoscopies were performed, which revealed multiple superficial mucosal lesions of the duodenum and active oozing of blood from the ileocecal junction (), and the biopsy specimen revealed lymphocytic duodenitis, which was consistent with GVHD. The problem ahead of us was controlling the severe GI hemorrhage of a patient with RBC counts less than 3×10/L, hemoglobin level less than 60 g/L, and platelet counts less than 30×10/L, with a safe method. After careful consideration, we made a bold attempt to introduce the technique of SAE for the treatment of this patient.
The procedure was performed in an angiography room under fluoroscopic guidance. Using local anesthetics, a retrograde catheterization of the left femoral artery was carried out in order to perform digital angiography of the aortoiliac sector, by modified Seldinger’s technique. Subsequently, selective catheterization of the celiac artery, with a standard angiographic catheter (Cobra 5F), was carried out to localize the bleeding vessels. Then, the catheter was guided into the superior mesenteric artery at the initial segment of the pancreaticoduodenal artery, where digital subtraction angiography (DSA) showed obvious signs of contrast extravasation, which was confirmed as the bleeding site (). A Progreat® microcatheter was guided into the bleeding branch, and DSA confirmed the previous finding again; then 700 to 1,000 µm gelatin sponge microparticles were used to occlude the bleeding branch of the pancreaticoduodenal artery until blood flow was ceased. Subsequently, a further angiography was performed to confirm the occlusion of the bleeding branches (). The catheter was removed after verifying that there were no other bleeding sites. Action of the unilateral lower limb was prohibited for over 12 hours after the angiography.
The patient’s GI bleeding stopped following the arterial embolotherapy; there was no further bleeding at the site of embolism and also no clinically obvious bowl necrosis. Two days later, a fecal occult blood test was performed, and the result was negative. The counts of RBCs and hemoglobin also increased gradually, while bloody diarrhea and related symptoms had ceased after treatment and had no recurrence. Though GVHD still existed, the GI hemorrhage had been fully controlled.


A 68-year-old female was referred to the clinic of Oral and Maxillofacial Radiology for CBCT preparation, as pre-operative imaging for implant insertion in the maxilla. She looked healthy without any history of sinus disease such as headaches, nasal discharge, facial pain and voice abnormalities, hyposmia or anosmia, and purulent rhinorrhea. On physical examination, it was found that the right maxillary region was slightly depressed compared with the opposite side. She did not remember any history of infection or trauma in the right maxillary sinus. CBCT was performed by a Gallileos set (Sirona dental systems GmbH, Bensheim, Hessen, Germany), and analyzed by Sidexis-XG software with slices having an interval of 2 mm. Evaluation of coronal and axial views revealed that the maxillary sinus of the right side was completely absent in all of the cuts. Ethmoid, sphenoid, and frontal sinuses were normal [].
Due to the lack of any symptoms, no additional treatment was done and the patient was informed about the sinus condition for possible future symptoms.


A 20-year-old female was admitted to the clinic of Radiology for CBCT of the maxilla as pre-operative diagnostic imaging for implant insertion. She was completely healthy without any history of headaches, nasal discharge, facial pain, voice abnormalities, hyposmia or anosmia, and purulent rhinorrhea. Also, she had no history of infection or trauma in the sinus areas. CBCT was performed and evaluation of coronal and axial views revealed that all the left sinus dimensions were less than those of right sinus, and its dimensions seemed to be only equal to half of those of right sinus [].
Due to the lack of any symptoms, no additional treatment was done and the patient was informed about the sinus condition for possible future symptoms.


A 12-year-old female was referred to the clinic of Dentomaxillofacial Radiology by her orthodontist for CBCT of both mandible and maxilla. She had a history of cleft palate and lip that had been treated surgically. She suffered from chronic headaches, nasal discharges, and voice abnormality because of hypernasalism. On physical examination, the scar of previous surgery was seen in the philtrom region. Also, the maxillary sinus regions in both sides were depressed. CBCT was performed and evaluation of coronal view revealed that the maxillary sinuses in both sides were absent in all slices, which was confirmed by the axial view as a bilateral aplasia of the maxillary sinuses [].
The patient was informed about the sinus condition and was refered to an otolaryngologist for further assessment.


A 24-year-old pregnant woman (gravida 1, para 0) whose medical and familial histories were unremarkable, presented with sore throat and a palpable mass in the right submandibular area. Neck biopsy of the submandibular mass revealed DCBCL, a subgroup of NHL.
She was at 22 weeks' gestation at the time of diagnosis and was recommended for termination of pregnancy at a different hospital. But we explained the risk of chemotherapy in pregnancy to the patient and her family before chemotherapy and the patient and her family agreed to receive the chemotherapy during pregnancy. Subsequently, the patient underwent neck and abdominal magnetic resonance imaging (MRI) (). A 2.4×1.7-cm enlarged lymph node at the right level IIA () and a 5.4×2.5-cm mass in the right palatine tonsil () were identified in neck MRI images. Initial blood tests revealed normal blood counts and chemistry, including a normal beta-microglobulin level of 1.16 mg/L. The fetal weight was estimated to be approximately 1,582 g at 29 weeks' gestation on ultrasonography. The fetus showed no abnormal findings. The patient was treated with R-CHOP chemotherapy immediately after evaluation for reproductive toxicology and receiving approval from the hematologists and the patient. The patient and fetus were closely monitored before and after induction of chemotherapy.
The patient received the standard dose of R-CHOP every 3 weeks: 375 mg/m2 rituximab, 750 mg/m2 cyclophosphamide, 50 mg/m2 doxorubicin, 1.4 mg/m2 vincristine, and 100 mg/day prednisone on days 1 to 5. A total of 6 cycles were planned. On the first day of chemotherapy, the patient complained of mild skin rash following rituximab infusion, which resolved following an injection of hydrocortisone. After the second cycle of chemotherapy, she had preterm contractions, which resolved following treatment with tocolytics (magnesium sulfate) for several days. Follow-up neck MRI showed marked reduction and almost complete disappearance of homogenous masses both in the neck () and right tonsil ().
After 4 cycles of R-CHOP, she was admitted to the hospital with preterm contraction at 33 weeks and 2 days' gestation. Nitrazine and premature rupture of membrane tests were negative, and the fetal estimated weight and other fetal recordings were within normal range. Emergency cesarean section was performed at 34 weeks and 5 days' gestation because of fetal distress (decreased variability:recurrent variable deceleration).
A 2,560 g, healthy baby girl was born with an APGAR (activity, pulse, grimace, appearance, and respiration) score of 7/9 without any complications. Echocardiography, brain ultrasonography, blood tests, whole body radiography were performed on the infant on the second day. Echocardiography showed a 1.2 mm patent ductus arteriosus, which was closed the following day. All other test results were unremarkable.
After delivery, the patient was administered the remaining 2 cycles of R-CHOP therapy. The follow-up computed tomography and, positron emission tomography/computed tomography scans showed complete remission. At 13 months after delivery, the patient showed no evidence of lymphoma, and her child weighed over 10 kg and did not show any developmental delays or physical abnormalities.


A 36-year-old, nulligravida, unwanted for pregnancy was referred to our hospital due to persistent dysmenorrhea and lower back pain following a bilateral uterine artery embolization and intrauterine device (Mirena) insertion at a local clinic. A pelvic magnetic resonance imaging showed a 5-cm-heterogeneous mass on the posterior uterine wall, which appeared to be an intramural fibroid. After thorough counseling on the potential risks and benefits of the procedure and the possible alternative surgical treatments was the laparoscopic ultrasound-guided RFA performed under general anesthesia. A dense adhesion was found in the right cul-de-sac between uterus and colon. Adhesiolysis and biopsies were performed of the adhesive band and peritoneum and an endometriosis was later confirmed. A 16-gauge RFA needle was percutaneously inserted and placed within the target fibroid under laparoscopic video and transvaginal ultrasound guidance. The RF delivery system (M1004, RF Medical Co., Seoul, Korea) was applied twice with a maximum power of 120 watt and a maximum temperature of 85℃ for 5 minutes. The patient was discharged without any complications. However, she visited the hospital for bloody anal discharge and vaginal leakage of stool after two weeks of discharge. During her visit were the vital signs stable, but a leukocytosis (white blood cell 12,820/µL) and an elevation of C-reactive protein (1.4 mg/dL) were observed. The colonoscopy showed a white, marginal protruding mucosal defect about 9 cm above the anal verge (). A pelvic computed tomography and a hysterosalpingogram revealed a fistula between the posterior uterine wall and the rectum (). The patient underwent a rectal fistula wedge resection and ileostomy and total abdominal hysterectomy due to severe inflammation and necrosis in the uterine cavity. Two months later, the fluoroscopic examination of the pouchgram showed an improved rectouterine fistula and no leakage of contrast media, thus a take-down of the ileostomy was performed. There was no further clinical problem afterward for one year of follow-up.


A 20-year-old male was admitted with 20 days history of fever and reddish lesions on the chest which rapidly progressed to involve the entire body over a period of 14 days. Clinical examination revealed papules, plaques, vesiculobullous and few ulcerative lesions on the chest, back, extremities, palms, soles, and genital mucosa [].
On general physical examination patient looked toxic with tachycardia (heart rate = 124/min), tachypnea (respiratory rate 22/min), fever (max. temp. 103.8 F), no lymphadenopathy was noted. Systemic examination revealed no abnormality. Differential diagnosis of vasculitis, erythema multiforme major, FUMHD, and pemphigus foliaceus were considered.
Histopathological examination from papulovesicular lesion revealed epidermal clefts, edema, and vacuolar degeneration of keratinocytes, basal cell degeneration, and dermal perivascular lymphocytic infiltrate without extravasation of RBC []. There was transient leucocytosis (TLC-12,300) and elevation of transaminases (serum glutamic oxaloacetic transaminase (SGOT)-50, serum glutamic pyruvic transaminase (SGPT)-63) which returned to normal over a period of 2 weeks. Direct immunoflouroscence, Veneral Disease Research Laboratory test (VDRL), enzyme-linked immunosorbent assay (ELISA) for human immunodeficiency virus (HIV), serology for herpes 1 and 2, Tzanck smear, X-ray chest, and ultrasonography of abdomen were normal. On clinical and histopathological basis, he was diagnosed as a case of FUMHD.
There was an initial good response to oral prednisolone 60 mg (1 mg/kg) and oral doxycycline 100 mg twice daily. His steroid was tapered by 10 mg weekly. However, there was a relapse on reducing steroids from 60 to 25 mg over 4 weeks. Methotrexate was added at this stage starting at 7.5 mg gradually increased to 15 mg in 2 weeks, which led to a dramatic response and it was possible to discontinue steroids and doxycycline in another 3 weeks. Patient is in complete remission on maintenance dose of 5 mg methotrexate since past 3 months. It is planned to continue methotrexate in the same dose for another 3 months and then continue follow up for 1 year without treatment. The papules and plaques healed with post-inflammatory hyperpigmentation (PIH) whereas vesiculobullous lesions with varioliform scarring [].


A 42-year-old man presented with a severe cough over a period of 2 months. His past medical history included pulmonary tuberculosis for which treatment was completed 1 year prior and diabetes mellitus, which was treated with insulin for over 14 years.
On admission, he had a severe cough, unexpectorated sputum, and chest discomfort. All his vital signs were stable except for a low-grade fever. He had a very thin physique and a chronic ill-looking appearance. Chest auscultation revealed fine crackle sounds in the left lung fields.
Laboratory work-up revealed the following: leukocyte count, 12,160 cells/mm3 (neutrophils 75%, lymphocytes 16.5%, monocytes 5.7%, eosinophils 2.5%, and basophils 0.2%); hemoglobin level, 12.3 g/dL; glycosylated hemoglobin, 10.1%; platelet count, 422,000 cells/µL; C-reactive protein level, 3.95 mg/dL; and pro-calcitonin quantitative level, <0.05. The other blood chemistry values were within normal limits, and the human immunodeficiency virus test and serum galactomannan index were negative. Three sputum acid-fast bacilli (AFB) tests were also negative.
A chest radiograph revealed a destroyed tuberculosis scar in the left and right middle lung fields, and this finding showed no interval change compared to that in a chest radiograph obtained a year ago (). A chest computed tomography scan showed a fibrotic cavity, traction bronchiectasis, and multiple small nodules with destructive changes in both lungs ().
Empirical antibiotic therapy was initiated immediately on suspicion of community-acquired pneumonia; the patient did not show any improvement with this treatment.
On the seventh hospital day, the patient underwent a flexible bronchoscopy for an evaluation of causality. The bronchoscopic examination demonstrated multiple, discrete, gelatinous whitish plaques involving mainly the trachea and left bronchus (). We performed a biopsy of the tracheal lesions, which revealed only chronic active inflammation with necrotic tissues. The AFB, periodic acid-Schiff, and Grocott's methenamine silver stains of the biopsy tissue were all negative.
The patient's symptoms did not improve in spite of empirical antibiotic treatment. On the 14th hospital day, he underwent a second bronchoscopic examination. There was no interval change compared to previous bronchoscopic findings. Another biopsy performed at this time revealed the same findings as the previous biopsy. Although the pathology was not confirmed, we made a presumptive diagnosis of Aspergillus tracheobronchitis on the basis of his symptoms, discriminative bronchoscopic findings, and unresponsiveness to antibiotics. On the 16th hospital day, empirical antifungal therapy with intravenous amphotericin B was initiated, and antibiotic administration was discontinued. However, 5 days later, we changed the drug to itraconazole owing to drug fever and renal insufficiency related to amphotericin B administration. After 7 days of intravenous itraconazole administration, the intractable cough, unexpectorated sputum, and chest discomfort symptoms started showing improvement. On the 35th hospital day, he underwent a follow-up bronchoscopic examination, which revealed a significant improvement in the numerous gelatinous whitish plaques in the trachea and left bronchus (). On the 44th hospital day, the patient was discharged on oral itraconazole with significant improvement of his symptoms. After 2 months, the Aspergillus species was isolated upon microbiological assessments of the sputum, washing fluid, and biopsy specimens. We finally confirmed the diagnosis of Aspergillus tracheobronchitis and continued treatment with oral itraconazole for 6 months. Oral itraconazole was discontinued after the patient demonstrated clinical improvement, bronchoscopic resolution, and confirmation of microbiologic eradication.


A 51-year-old postmenopausal woman presented with frontal hair thinning occurring during 3 years. Four years before her presentation, she underwent modified radical mastectomy, chemotherapy (doxorubicin and cyclophosphamide), and radiotherapy (cumulative dose, 5,040 cGy) for her breast cancer. While receiving the chemotherapy, she showed total hair loss on the scalp, suggestive of anagen effluvium, from which she fully recovered after several months. To prevent the recurrence of the cancer, further hormonal anticancer therapy with selective estrogen receptor modulators (SERMs) (toremifene citrate, Fareston; Prostraka Inc., Somervillle, NJ, USA) was subsequently initiated. Two to three months later, she developed hair thinning limited to the frontal and parietal scalp. Dermatological examination showed typical male pattern alopecia with moderate fronto-parietal hair thinning and recession of the frontal hairline (). The hairs of the temporal and occipital scalp, as well as other body hairs, were normal. She had a family history of androgenetic alopecia on both paternal and maternal sides. She was treated with 3% minoxidil, 0.025% alfatradiol, and 0.025% tretinoin solution twice daily. Spironolactone (200 mg/d) and finasteride (1 mg/d) were added to the regimen at 1 month and 3 months after the initial treatment, respectively. After 4 months of follow-up, improvement in hair density and hair diameter in the fronto-parietal scalp was observed.


A 33-year-old premenopausal woman presented with a 1-year history of fronto-parietal hair thinning. Four years before presentation, she had undergone total mastectomy and chemotherapy with doxorubicin, cyclophosphamide, docetaxel, and tegafur-uracil. While receiving the chemotherapy, she showed total hair loss on the scalp and body, from which she fully recovered after several months. From 1 year before the presentation, she had undergone hormonal anticancer therapy with SERMs (tamoxifen citrate, Nolvadex; AstraZeneca Pharmaceuticals LP, Wilmington, DE, USA). One month later, she noted hair thinning on the top of her scalp. There was no family history of alopecia. Dermatological examination showed decreased hair density and hair diameter in the frontal and parietal scalp. She was treated with 3% minoxidil, 0.025% alfatradiol, and 0.025% tretinoin solution twice daily for the alopecia; however, she refused to continue the treatment.


A 51-year-old postmenopausal woman presented with a 6-month history of fronto-parietal hair thinning. Three years before presentation, she had undergone wide local excision and axillary lymph node dissection, chemotherapy (doxorubicin, cyclophosphamide), and radiotherapy (cumulative dose, 6,000 cGy) for breast cancer. Additionally, after finishing all those treatments, she took aromatase inhibitors (AIs) (anastrozole, Arimidex; AstraZeneca Pharmaceuticals LP) for 9 months before her visit to our clinic. The hair loss that developed initially was recovered but the fronto-parietal hair thinning persisted. She had a family history of androgenetic alopecia on the paternal side. On dermatological examination, she had frontal recession with decreased hair density and hair diameter limited to the frontal and parietal scalp. She was subsequently treated with 3% minoxidil and 0.025% alfatradiol solution twice daily. After 6 months, a slight improvement in hair density and hair diameter in the frontal and parietal scalp was observed.


A 50-year-old postmenopausal woman visited our dermatology clinic complaining of hair thinning on the fronto-parietal scalp occurring during the 10 months before her visit. Two years before presentation, she had undergone modified radical mastectomy, chemotherapy (doxorubicin, cyclophosphamide, and docetaxel), and radiotherapy (cumulative dose, 5,040 cGy) for breast cancer. Then, AIs (anastrozole, Arimidex) were subsequently administered to prevent the recurrence of the cancer. She had a family history of androgenetic alopecia on the paternal side. Dermatological examination revealed female pattern alopecia showing diffuse hair thinning limited to the fronto-parietal scalp with a preserved frontal hairline (). She was prescribed with 3% minoxidil and 0.025% alfatradiol solution twice daily. However, she wanted treatment only for her primary concern (i.e., breast cancer), and discontinued the dermatological care.


A 50-year-old postmenopausal woman presented with a 1-year history of scalp hair loss, particularly of the frontal and parietal scalp. Six years before presentation, she had undergone breast cancer treatment that was similar to that of case 4. To prevent recurrence, SERMs (toremifene citrate, Fareston; tamoxifen citrate, Nolvadex) were administered for up to 1 year before her visit to our clinic. Switching from SERMs, she was subsequently given AIs (letrozole, Femara; Novartis Pharmaceuticals Corp., East Hanover, NJ, USA). Two months after those medications, she developed hair thinning limited to the parietal scalp without recession of the frontal hairline. She reported no family history of hair disorders. She was treated with 5% minoxidil and 0.025% alfatradiol solution twice daily, and slight improvement in hair thickness was observed after 3 months of follow-up.


A 52-year-old, right-handed, Caucasian male was referred to our outpatient neurosurgery clinic for evaluation of progressively worsening headaches over a 2-week period. On examination, the patient did not have exophthalmoses, chemosis, or bruit, and was otherwise neurologically intact. There was, however, a prominent right superficial temporal artery that engorged with valsalva-type maneuvers.
Cerebral angiography revealed a dAVF in the anterior cranial fossa. The fistula was fed by bilateral ophthalmic arteries via the anterior ethmoidal branches [Figure and ], as well as a frontal branch of the right superficial temporal artery []. The fistula drained to the superior sagittal sinus via cortical veins with associated venous varix dilatation (Cognard type IV lesion). Injection of the left ICA also revealed a large, wide-necked, 10-mm carotid-ophthalmic aneurysm [] incorporating the parent artery [Figure and ]. Given the angioarchitecture of the carotid-ophthalmic aneurysm, microsurgical obliteration of the fistula and the aneurysm was undertaken instead of any endovascular treatment to minimize the risk of ophthalmic artery occlusion and blindness.
The patient's dAVF was successfully obliterated using standard microsurgical techniques through a right pterional craniotomy. Clipping of the contralateral carotid-ophthalmic aneurysm was attempted but since it required significant brain retraction, an intraoperative decision was made to abandon aneurysm clipping with the understanding that the patient could undergo coiling of the aneurysm shortly after surgery. Obliteration of the AV fistula was confirmed intraoperatively by fluorescein angiography. The patient was discharged home after an uneventful hospital course and a short-term, follow-up visit revealed resolution of the patient's headaches.
The scheduled postoperative cerebral angiography with intent to coil the carotid-ophthalmic aneurysm a mere 28 days following surgery revealed no fistulous connection, return of bilateral ophthalmic arteries to their normal caliber, and an 85% reduction in size of the aneurysm. The small, residual aneurysm appeared round with a smooth, regular outline []. Because of this dramatic reduction in size of the aneurysm and its benign angiographic morphology, we elected not to coil the aneurysm and instead opted for conservative management with serial imaging and aggressive follow-up.


A 15-year-old girl presented to our pulmonology outpatient department with a long history of chronic productive cough (clear sputum) and shortness of breath on exertion. She was able to comfortably perform activities of daily living, but complained of being unable to participate in school sports because of cough and dyspnea. Her background medical history included vertically acquired HIV infection for which she had been treated with ART since age 3 years. She had two previous episodes of fully treated, culture-confirmed, drug-sensitive pulmonary tuberculosis, both before the age of 5 years. There had also been three documented admissions for severe pneumonia in childhood. She had been switched to second-line ART two years previously for virological and immunological failure secondary to nonadherence, coinciding with a time when her HIV-infected mother was also ill and unable to supervise her treatment.
Physical examination revealed digital clubbing, hyperinflation and reduced breath sounds with mild wheeze globally, and crackles in the lower zones. There was no clinical or electrocardiographic evidence of pulmonary hypertension. Her growth chart showed decreased growth velocity; both height-for-age and weight-for-age were on the third centile. Spirometry showed marked airflow limitation (forced expiratory volume in one second (FEV1) 33% predicted) with nonsignificant bronchodilator reversibility, and a moderate-to-severe impairment in diffusing capacity (53% predicted).
The chest radiograph showed hyperlucency of the left lung with multiple cystic shadows in the lower zones bilaterally (Fig. ). The high-resolution computed tomography of the chest confirmed bilateral cylindrical and saccular bronchiectasis, with mosaic attenuation and gas trapping on expiratory views (Fig. ). She had a recent CD4 count of 406 cells/milliliter and a suppressed HIV viral load. Sputum cultured a sensitive H. influenzae; Xpert® (Cepheid Inc., Sunnyvale, CA, USA) MTB/RIF and culture for M. tuberculosis were negative.
She was prescribed inhaled corticosteroids and long-acting beta-agonists and azithromycin three times a week (on alternate days) on a continuous basis. She was given the polysaccharide pneumococcal and influenza vaccines. In the subsequent visits in the 12 months of follow-up so far, her shortness of breath has subjectively improved, but there has been no appreciable improvement in her lung function. She has had no further infective exacerbations.


An 81-year-old male patient was treated for prostatic cancer with radiotherapy (total dose 71 Gy) 4 years ago. He presented with repeated episodes of rectal bleeding and a hematocrit of 22%. A rectosigmoidoscopy showed telangiectasia of his rectum, consistent with features of radiation proctopathy ().
He went through one session of RFA and did very well after the treatment, with stabilization of his hematocrit (). The ablation started in the distal rectum (spanning approximately 5 cm), with a total procedure time of approximately 12 minutes. However, 2 months later, he developed deep vein thrombosis and had to be started on warfarin. Six weeks after this he developed a gastrointestinal bleed again.
Repeat rectosigmoidoscopy showed a lot of improvement in the telangiectasia and his rectal mucosa, but a second session of RFA, 3 months after the initial one, was done to prevent further bleeding (). The second ablation also started in the distal rectum, spanning 5 cm, with a total procedure time of 12 minutes.
His hematocrit has improved and has remained stable after the procedure. We have followed this patient up for 6 months since the first procedure. The procedure required a gastroscope for better maneuverability in the rectum to perform RFA, with a Halo-90 paddle demanding 12 J of energy. Another sigmoidoscopy done 6 months later showed marked improvement in the mucosal ectasia.


An 83-year-old male patient with a history of prostate cancer treated with external-beam radiotherapy (total dose 74.4 Gy) in 2008 presented with rectal bleeding. His medical history was also significant for coronary artery disease with stent placement, requiring him to be on clopidogrel and aspirin. His hematocrit on presentation was 20%. Rectosigmoidoscopy showed rectal disease consistent with radiation proctopathy ().


A third case, an 81-year-old male patient, with a history of prostatic cancer diagnosed in 2009, presented with rectal bleeding. He was treated with external-beam radiotherapy (total dose 77.3 Gy) in 2009 and presented with multiple episodes of rectal bleeding and a hematocrit of 21%. His medical history was significant for Parkinson’s disease and cerebrovascular accidents in 2006 and 2010, requiring him to be on aspirin. His rectosigmoidoscopy revealed significant telangiectasia consistent with CRP ().
He was treated with RFA using a Halo-90 catheter at 12 J energy setting. The ablation started in the distal rectum (spanning approximately 8 cm), with a total procedure time of approximately 20 minutes. The patient has done well since the procedure, with no further bleeding reported and a stable hematocrit of 28%–30% ().


A 68-year-old woman presented with chronic non-healing ulcers above the waistline on both flanks (lady was tying her saree with petticoat at this level) with duration of 8 months. Rapid progression of the ulcer on left flank with growth like lesion had been noticed since past 2 months. Both the ulcers were preceded by hypopigmented patches and chronic itching at the site for some years. She has been wearing saree since the age of 13 yrs. On clinical examination, an ulceroproliferative growth measuring 2 cm × 2.5 cm, with surrounding well-defined hypopigmented margin, was found on her left loin. Serosanguinous discharge was present from the lesion. On palpation, the ulcerproliferative growth was tender and was not fixed to the underlying structures. The surrounding skin was scaly. An ulcer measuring 2.5 cm × 5 cm with everted edge and surrounded by area of hypo and hyperpigmentation with induration was present on the right side of the waist []. Bilateral inguinal lymph nodes were multiple, nontender, discrete, firm and mobile. A wedge biopsy from both lesions showed a well differentiated SCC. Complete metastatic workup was done and was negative. The patient had no co-morbidities. Surgery was planned and wide excision of the growth with 2 cm margin and rhomboid transposition flaps on both sides was done []. On histopathology, the tumour was found to be composed of a hyperplastic epidermis with sheets of keratinocytes (with intercellular bridges) demonstrating intense mitotic activity, pleomorphism, and greatly enlarged hyperchromatic nuclei and scanty cytoplasm, suggestive of a well-differentiated SCC with free resection margin of at least 8 mm on all sides. Post-operative period was uneventful [Figures and ]. Inguinal lymphadenopathy resolved after an antibiotic course. Follow-up was done up to 8 months during which the patient remained disease free.


A 53-year-old woman visited our hospital complaining of intermittent nocturnal anterior chest pain for several months. Her medical history was non-contributory. Initial contrast-enhanced chest CT revealed several, variable-sized, oval to lobular, relatively circumscribed, well-enhanced masses in both breasts, with an enlarged right internal mammary lymph node () and an enhanced pleural mass in the left hemithorax (not shown). The initial impression of these lesions was multifocal bilateral (cancer) metastases to the breast of unknown origin with pleural and right internal mammary lymph node metastases. An US evaluation of both breasts showed that the largest mass in each breast was oval, with an indistinct margin and heterogeneous echogenicity (, top and middle) with posterior acoustic enhancement at 2 o’clock in the left breast and 10 o’clock in the right breast. Color Doppler US showed that the masses had internal vascularity (, bottom). These masses were classified as category 4 A (low suspicion for malignancy) according to the Breast Imaging Reporting and Data System (BI-RADS) for US. Subsequently, the patient underwent US-guided core needle biopsies of both breasts. On immunohistochemical staining, the tissue was positive for CD79a, a B cell marker (), IgG, and the lambda light chain (). Both breast masses were confirmed to be IgG lambda type plasma cell myeloma. A systemic evaluation of the patient revealed bone involvement in the spine, ribs, sternum, left scapula, sacrum, and both iliac bones on plain radiographs and CT. Therefore, the patient was diagnosed with secondary BP as an extramedullary presentation of MM. Accordingly, chemotherapy with thalidomide and dexamethasone was started 10 days after the initial US evaluation. On the day of chemotherapy initiation, mammography performed shortly after infusing the chemotherapeutic agents revealed multiple small circumscribed or indistinct margin, oval to round, bilateral breast masses (). US performed on the same day showed significant interval size reduction of the bilateral breast masses compared to the initial US (not shown). The largest lesion in the left breast at 2 o’clock direction had decreased from 5 cm to 3.5 cm in maximum diameter. On chest CT after 3 months (not shown), all of the breast lesions, the left pleural mass, and the right internal mammary lymph node showed a marked reduction in size.


A 14-year-old girl with an uneventful clinical history suddenly developed a severe syndrome characterized by intermittent-remittent fever with spikes up to 41℃, headache, meningismus, drowsiness, myalgia, and arthralgia without arthritis. A mild liver and spleen enlargement was present.
Routine investigations showed increased acute phase reactants (erythrocyte sedimentation rate [ESR], levels of 90 mm/h; C-reactive protein [CRP], 14.7 mg/dl), anemia [hemoglobin 9.6 g/dl], total white cell count 6.4 × 103/µL, thrombocytopenia [128 × 103/µL], and liver involvement [aspartate transaminase 322 UI/L, alanine transaminase 485 UI/L, and γ-GT 295 UI/L]). Serology for some hepatotropic viruses (i.e. hepatitis viruses A-B-C, Epstein-Barr virus, cytomegalovirus) was negative. The autoantibody panel showed a slight autoantibody positivity of anti-smooth muscle antibody (ASMA) (1:40) and perinuclear anti-neutrophil cytoplasmic antibodies (p-ANCA) with an atypical pattern.
Due to the increased transaminase and γ-GT plasma levels a liver biopsy was performed with the results being consistent with a non-specific hepatitis pattern. Moreover, brain MRI, cerebrospinal fluid analysis and bacterial culture ruled out encephalo-meningitis; bone marrow biopsy and bone scintigraphy were also negative. Antibiotics treatment (amoxicillin-clavulanate 90 mg/kg/day three times a day) was started without significant clinical response. Since fever spikes and myalgia/arthralgia persisted, the girl was admitted to our department 30 days after disease onset.
At admission, the patient complained of recurrent fever spikes and severe malaise, although according to the parents’ opinion the general conditions of the girl had been ameliorated. Laboratory investigations showed positive acute phase reactants (ESR 75 mm/h and CRP 3.9 mg/dl), hypergammaglobulinemia (1.73 g/dl) with increased IgM (241 mg/dl) and IgA (490 mg/dl) plasma concentrations: notably, transaminase activities were normal with persistently increased γ-GT 114 UI/L. As a diagnosis had not yet been reached, we performed a WB-MRI on a 1.5 T MRI scanner (Achieva Intera; Philips Medical Systems, Best, The Netherlands) using body coil with a field of view covering from head to toes. WB-MRI images were acquired (and displayed) in coronal plane using a turbo spin echo T1-weighted (T1W) sequence (TR, 630 ms; TE, 17 ms; slice thickness, 5 mm; gap, 0.5 mm; NSA, 1) and a turbo spin echo STIR sequence (TR, 2800 ms; TE, 165 ms; TI, 64 ms; slice thickness, 5 mm; gap, 0.5 mm; NSA, 3). Total scan time was about 30 min. No contrast medium was administered during the examination.
The MR investigation was carried out with the aim to rule out hemato-oncologic diseases and occult abscess. Surprisingly the WB-MRI showed an unusual pattern characterized by millimetric, widespread punctate nodules, respectively hypo-hyperintense in T1W and STIR sequences. These nodules were mainly localized in the meta-epiphyseal regions of the long bones of both legs (mostly at the level of the distal femur and proximal tibia) as further documented by an additional dedicated MRI of both knees (, , , and ). According to the radiologist’s opinion, these images were possibly related to a hematologic intramedullary spread of an infectious process or to a post-infectious thrombotic microangiopathy.
Upon this finding, and taking into account the apparent self-limiting disease course and the rural area where the patient lived, further investigations were conducted to rule out zoonosis. Serologic assay of anti-Anaplasma phagocytophilum showed an IgM titer of 1:40 (positive, >1:64) and an IgG titer of 1:160 (positive, >1:64). Hence intravenous ceftriaxone treatment (80 mg/kg daily) was started with a prompt clinical improvement (i.e. disappearance of fever, myalgia, malaise, and headache). The patient has not had a relapse after about 1 year of treatment.