A 27-year old man presented to our outpatient department with increasing pain in the region of the right sacroiliac joint. The pain started three years earlier, at which point the patient sought help from a local orthopaedic surgeon. At this initial presentation it was thought that his pain was attributable to his physically demanding job. A CT-scan and MRI-scan were performed and revealed a bone cyst with sclerotic borders without destruction of the cortical bone (Figs. , ). Signs of malignancy were not present. Accordingly, the patient was treated with physical therapy, chiropractic treatment, and pain control.
Over time his pain increased to the point of requiring opioids for pain control and presenting to our hospital. In the interim, he changed his job and decreased his physical activity, neither of which alleviated his pain.
On physical examination the patient had tenderness to palpation and vibration over his right sacroiliac joint. Peripheral motor and sensory functions were intact, no pathologic reflexes were present. The patient was in a good general state of health. A CT-scan was repeated and showed a slightly enlarged bone cyst compared to the previous CT-scan three years ago. However, no cortical breakthrough was present (Fig. ).
For further diagnostic workup an open biopsy of the lesion was performed one week later. The histological examination of the tissue demonstrated the formation of abundant collagen fibers and spindle shaped cells with medium to low cell density. The nuclei were without pleomorphism and mitotic activity. In immuno-staining the nuclei of the tumor cells were positively labeled for the adhesion protein β-catenin. In addition there was a partially positive staining recognized for S-100. Morphology and immunophenotype were consistent with either aggressive fibromatosis or desmoplastic fibroma, both of which express β-catenin (Fig. ). Because of its intraosseus location the working diagnosis of desmoplastic fibroma was made. Accordingly the indication for operative excision via curettage and grafting was established.
The operation was performed with the patient under general anaesthesia. The tumor was removed in several parts. The tissue was beige in color with hard and soft areas of consistency (Fig. ). The removed tumor was sent for further histological examination to the department of pathology.
The lesion was filled with autologous bone graft from the iliac crest that was mixed with Cerasorb® (Curasan, Kleinostheim, Germany) and blood from the patient. Operating time was 105 minutes. After surgery the patient was restricted to no weight bearing on the operated side until complete wound healing. Compared to a more invasive treatment option, this therapeutic approach has the advantage of reduced operating time and earlier recovery.
No postoperative radiation of the lesion was performed to enable adequate integration of the bone graft.
Histological workup of the removed tissue demonstrated the identical picture as the biopsy and accordingly no signs of malignancy.
Postoperative follow-up examinations as well as CT-scans 6 and 18 months after surgery showed good integration of the graft (Figs. , ). There were no signs of recurrence.


The patient was a 24-year-old male who presented with dyspnea. He was cyanotic, and the oxygen saturation in the room air was 65%. The patient had clubbing in his toes and fingers. No procedure had been performed for this patient until then.
Echocardiography revealed moderate enlargement and normal function of a predominant morphologic right ventricle (RV). Both of the atrioventricular (AV) valves opened to the RV with mild regurgitation, and both great arteries arose from the RV with severe subvalvular and valvular pulmonary stenosis. The position of the great arteries was D-malposed; i.e. the aorta was anterior and right-sided.
The patient’s ventricular function rendered him amenable to the Fontan surgery. Catheterization measured the mean pulmonary artery pressure (PAP) and left ventricular end diastolic pressure (LVEDP) as 10 mm Hg and 12 mm Hg, respectively. During RV injection, it was observed that the left pulmonary artery (LPA) was not opacified; decision was, therefore, made to conduct magnetic resonance imaging (MRI) so as to better evaluate the pulmonary artery branches. The procedure showed a long, significant stenosis of the LPA, which was 25 mm in length and 4 mm in diameter ( and ).
Given the posterior position of the LPA and the relative distant location of the stenosis from the bifurcation and the desire to reduce surgery time, a hybrid LPA stenting procedure was opted for. After median sternotomy and routine cannulation, cardiopulmonary bypass was performed, the heart was arrested, and the Glenn Shunt was conducted on the beating heart with low-grade hypothermia and without an aortic clamp. Next, the main of the pulmonary artery (PA) was incised before the bifurcation, and a 3- mm Bare Stent (Genesis, Cordis Co.) was mounted on a 12 × 40-mm balloon (Opta Pro, Cordis Co.), which was passed through the LPA. The course of the catheter and stent as well as the covering of the lesion with the stent was monitored via transesophageal echocardiography (TEE). The acceptable position of the stent having been ascertained, the balloon was inflated up to 5 Atm and the final diameter of the stent reached 12 mm. TEE confirmed the correct position of the stent and the diameter of the LPA after stenting (). After the procedure, oxygen saturation rose to 83%.
At six months’ follow-up of the patient, exercise capacity and cyanosis had improved significantly, with O2 saturation having reached near 85% by pulse oximetry. Furthermore, no restenosis was seen in the follow-up imaging by transthoracic echocardiography ().


This 11-year-old boy presented with a 3-year history of right buttock pain that radiated into his lower back and into his right lateral thigh. The pain was present most of the time regardless of activity such as walking or sitting. An initial workup involving magnetic resonance imaging (MRI) of the lumbar spine was unrevealing. The patient had significant atrophy of the right thigh, calf, and foot with an asymmetry in his shoe size with the right foot being three US shoe sizes smaller than the right. In December 2010, the patient presented to neurosurgical attention with progressive complaints of proximal right lower extremity pain, muscle spasms, and gait difficulty.
The patient's examination revealed a significant asymmetry in size as the measured circumference at the distal thigh was 53.5 cm at the left thigh versus 50.5 cm in the right thigh, 43 cm in the left calf versus 42 cm in the right calf. The length of his foot from the great toe to the heel was 27 cm in the left foot and 24 cm on the right foot. He had normal strength in the left lower extremity. His right lower extremity exhibited 5−/5 strength in the right hip flexor, 4+ in the quadriceps, 4+ in the hamstrings, 4+ in the hip abductor, 4+ in the hip adductor, 5− in the tibialis anterior, 4+ to 5− in the gastrocnemius, 5− in the extensor hallucis longus, 4+ in foot eversion, and 5− in foot inversion. He was unable to perform heel rise or toe rise on his right leg. He had slightly decreased sensation to light touch and pinprick in the posterior and medial portions of his right thigh, lateral aspect of the right leg, lateral right foot, second toe dorsally and fifth toe dorsally. The corresponding toes were noted to have nonpainful dorsal calluses []. The patient had negative straight leg raise bilaterally. Deep tendon reflexes were rated 2+ throughout except for an absent right ankle jerk reflex. Range of motion was full and symmetric in all lower extremity joints.
In addition, the patient exhibited tenderness to palpation in the infragluteal area in the right hamstring without radiation of pain, along with point tenderness in the region of the right knee lateral to the patella proximal to the head of the fibula. When walking, he complained of posterior thigh pain. The patient had no skin lesions overlying the right posterior thigh or elsewhere on his body, and no stigmata of neurofibromatosis. He had no scoliosis.
MRI studies revealed a lesion at the right sciatic nerve extending 30 cm from the sciatic notch to the distal thigh. The tumor appeared hypointense on T2-weighted images, isointense-to-hypointense signal on the T1-weighted image with mild heterogeneous internal enhancement in a longitudinal orientation after administration of gadolinium contrast []. Associated muscle atrophy in the sciatic nerve distribution was also noted. MRI of the pelvis demonstrated no extension superior to the sciatic notch.
The patient's electromyography (EMG) showed electrodiagnostic evidence of a right sciatic neuropathy with evidence of ongoing denervation and reinnervation as demonstrated by increased insertional activity, positive sharp waves, and fibrillation potentials in the distribution of muscles innervated by the sciatic nerve.
Given the patient's history and the need for tissue diagnosis to elucidate treatment options, the patient initially underwent an ultrasound-guided biopsy followed by a computed tomography (CT)-guided biopsy of the mass, both of which were unsuccessful at obtaining adequate tissue for histologic diagnosis. The patient underwent an open biopsy through exposure of the sciatic nerve via a posterior gluteus maximus-splitting approach. The whole sciatic nerve appeared involved and was notably hypertrophic, firm, and bulbous []. The epineurium was thick. Neurolysis revealed no visual evidence of intervening nonneural tissue among the large and fibrous fascicles. Two fascicles were selected for biopsy. Postoperatively, the patient had no new neurological deficits, and remained stable by subjective report and by clinical examination.
Pathological examination showed the lesion to be a neuromuscular hamartoma (neuromuscular choristoma). Within the specimen, small nerve bundles and axons were seen intermixed with adjacent mature striated skeletal muscle bundles, surrounded by a dense collagenous matrix []. The skeletal muscle was noted to have small, bland, peripherally placed nuclei with mature features and no cellular atypia or atypical mitoses.
The specimens were embedded in paraffin and the sections were stained with hematoxylin and eosin and trichrome. A panel of immunohistochemistry stains was also performed. The S-100 and neurofilament stains showed normal nerve bundles. The Epithelial Membrane Antigen (EMA) stain was inconclusive. Desmin highlighted the hamartomatous striated muscle fibers intermingled with nerve bundles, and myogenin stain was negative for immature skeletal muscular elements.


A 61-year-old woman without any risk factor of arteriosclerosis visited a nearby hospital because of mild head injury. Magnetic resonance (MR) imaging demonstrated no apparent traumatic lesion. However, flow-void sign of the right middle cerebral artery was sluggish []. Cerebrovascular disease was suspected and she was referred to our hospital for further examination. She was neurologically intact and had no significant history of weakness or sensory disturbance. Moreover, her family has not experienced cerebrovascular disease. Right carotid angiogram demonstrated stenosis of the terminal portion of the ICA with moyamoya vessels []. Left carotid angiogram and left vertebral angiogram showed no apparent abnormality [Figure and ]. Single photon emission computed tomography (SPECT) demonstrated hypoperfusion in the right cerebral hemisphere. The response to the acetazolamide was impaired []. She was diagnosed as unilateral MMD and followed by MR imaging annually. During 6 years, she had been intact and MR imaging had not detected progression of MMD. However, 7 years after the diagnosis, cerebral aneurysm appeared in the right basal cistern, probably from anterior choroidal artery, and conventional angiography was planed []. Before that, she become comatose and brought to our hospital emergently. Computed tomography (CT) showed massive intraventricular hemorrhage []. Enlargement of the aneurysm was revealed by CT angiography [Figure –]. Intracerebral hematoma was evacuated and the aneurysm was obliterated by bipolar coagulation. Postoperatively, she was in vegetative state and transferred to the rehabilitation hospital.


A 69-year-old male with history of chronic gastro-duodenitis and colonic diverticulosis was admitted to Emergency Department because of postprandial epigastric pain radiating to his left hemiabdomen, associated with vomiting. At physical examination, he was found dyspneic and tachypneic, and severe pain was exacerbated during palpation on the upper abdomen without signs of peritonism. Nasogastric tube positioning yielded enteric, partly coffee ground-like fluid.
Technically limited plain radiographs of the thorax and abdomen, obtained with the patient supine, reported predominantly left-sided basal lung opacities consistent with pneumonia and probable associated effusion. Shortly afterwards, because of worsening clinical condition and haemodynamic parameters, urgent contrast-enhanced multidetector CT was requested to rule out acute abnormalities of the thoraco-abdominal aorta.
Initial CT acquisition [] disclosed an abnormal posterior mediastinal air collection surrounding the collapsed distal esophagus, suggesting the clinically unsuspected presence of a perforation. Bilateral basal lung infiltrates, with extensive involvement of the left inferior lobe, were attributed to aspiration pneumonia. With the patient still on the CT scanner table, further investigation with CT-esophagography was performed with administration of diluted iodinated contrast medium through the nasogastric tube [Figure and ]. Evidence of extraluminal contrast leakage confirmed the presence of a focal wall disruption on the left postero-lateral aspect of the distal esophagus. Both the esophageal perforation and the associated mediastinal contamination were depicted along three planes by means of multiplanar CT reformations. Contrast-enhanced CT acquisition excluded acute aortic diseases.
Antibiotic treatment with intravenous piperacilline was started. Urgent surgery was performed, including thoracotomic access, posterior mediastinal debridement and irrigation. In agreement with CT findings, after a full-thickness distal esophageal perforation was identified and primarily repaired, without the need for resection. Drainage tube was left in place postoperatively. Supportive therapy and parenteral nutrition therapy were protracted during 32 days of Intensive Care Unit stay. Antibiotics were modified with introduction of cefazoline, according to pus cultures and antibiogram. The patient finally recovered and was discharged from the hospital nearly two months after initial admission.


A 28-year-old male presented with a single, right, level 2 cervical lymph node of 2 months duration. No other relevant history was elicited. Routine blood test showed normal total leukocyte count, with 25% eosinophils in the differential count. The lymph node excision specimen measured 2 cm in diameter, with a grey-white cut surface. Initial microscopic sections showed lymph node tissue with intense eosinophil infiltration in the paracortical, medullary and germinal center areas with focal eosinophil microabscess formation. There was prominent follicular hyperplasia. No parasites were seen in the initial sections [].
Lymph node eosinophilia elicits many provisional diagnoses like Kimura disease, Hodgkin disease, hypereosinophilic syndrome/leukemia, angiolymphoid hyperplasia with eosinophilia, parasitic infection etc. Many causes of lymph node eosinophilia mentioned above were ruled out because of the absence of specific findings. Kimura disease could not be excluded. Almost all the findings like marked eosinophil infiltrate, eosinophil microabscess, follicular hyperplasia, fitted well for the diagnosis of Kimura disease, and the residents offered this as a provisional diagnosis. But, on keen observation of the initial sections, one finding did not fit into Kimura disease – the presence of focal foreign body giant cell reaction against a pale eosinophilic glassy/hyaline material [Figures and ].
Even though eosinophilic material is described in Kimura disease, it is an amorphous type of eosinophilic proteinacious material[] without any giant cell reaction. The hyaline material led to the suspicion of a foreign body eliciting an eosinophil response and the patient being from a tropical country, the presence of a parasite was suspected.
Hence, the lymph node tissue was sectioned almost to the exhaustion of the wax block and a very careful search for an organism was made. In one or two serial sections, a few microfilariae were seen with a pale-staining ill-defined sheath and longitudinally arranged loose nuclei, the morphology suggesting microfilaria of filarial worm, possibly of Wuchereria bancrofti. The sheath is demarcated by adherence of inflammatory cells to its border, a finding also observed by Yenkeshwar et al, in their study.[] Thus, the thought about the possibility of a hidden parasite and the action of extensive sectioning and diligent search led to the correct diagnosis of the case [Figures –].


A 30-year-old female reported to orthopedic outpatient department with pain and swelling just above the right ankle joint and another swelling over the right iliac region for last 6 months. The patient had a surgery for the swelling of distal right leg 18 months ago by general orthopedic surgeon. Review of surgical records revealed that she was operated for a lytic lesion in distal tibia with preliminary diagnosis of benign bone lesion without preoperative biopsy. Curettage of the lesion was performed along with filling of surgical cavity with autologous bone grafts harvested from the right iliac crest. Histopathology revealed benign GCT of distal tibia. Below knee plaster was kept for 4 months postoperatively. Two months after the removal of plaster, she developed pain in the right leg just above the ankle, along with gradually increasing swelling. Two weeks later, she noticed another swelling over the right iliac crest, associated with mild pain and discomfort. The patient was finally referred to our oncologic clinic. The patient was clinically anemic. There was a swelling over the anterolateral aspect of distal part of right leg with painful restricted ankle movements and another swelling over the right iliac region with mild to moderate tenderness on palpation on both sites.
Plain X-rays showed lytic lesion in distal tibia with ill defined margins without any evidence of pathological fracture. X-ray of right iliac wing showed large soft tissue mass with irregularity of the bone over the outer table of iliac crest []. Magnetic resonance imaging (MRI) of pelvis and distal tibia was done to delineate the extent of tumor and its extension [Figures and ]. Technetium-99 bone scan showed increased uptake around distal tibia and focally in the ipsilateral iliac region only at the graft harvest site and no additional foci in the body (i.e. ruling out metachronous lesions elsewhere). Core needle biopsy was performed at both sites to confirm the microscopic/tissue diagnosis. The histopathology revealed benign GCT at both sites with identical picture. Computed tomography of the lungs was normal.
The patient was taken up for planned surgery under spinal anesthesia after taking informed consent. Distal tibial lesion was widely resected and reconstruction was done with medialization of fibula and fixing it on the dome of talus and in the distal medullary canal of the resected tibia with Kirscher's wires. Wide resection of iliac wing along with soft tissue mass was performed. The iliac swelling was confined to soft tissue underneath the scar with irregularity over iliac crest. The histopathology of both sites reconfirmed GCT.
Postoperative period was uneventful and the patient was discharged with a long leg cast for 4 months. Then, partial weight bearing was started with crutches. Patient achieved restoration of bone continuity with union at both ends. Hypertrophy of the grafted fibula was observed and patient was mobile with full unprotected weight bearing on the operated leg after 1 year. Two years after surgery, the patient was symptom free and walking without any aid and with no evidence of recurrence at both sites and no metachronous lesions or pulmonary metastasis. Fibula showed good hypertrophy and the iliac region healed well [].


A healthy 16-year-old boy was referred for the evaluation and management of an asymptomatic radiolucency of the left mandibular condyle that was noted on a routine panoramic radiograph. A history of no trauma was elicited, and the familial and medical histories were not contributory. Physical examination revealed a slight mandibular laterognathism, normal mandibular excursions, and a painless range of motion in spite of class III malocclusion. The occlusion was unstable, and clicking sounds were detected at the right temporomandibular joint (TMJ) during mouth opening. No cervical lymphadenopathy was detected, and no intraoral swelling or deformity was visible.
A panoramic radiograph showed a well-defined multilocular radiolucent lesion located in the left mandibular condyle. The mandible was deviated to the left side due to hypertrophy of the right mandible. Multiple lower anterior teeth were tilted to compensate for the hemimandibular hypertrophy, and the occlusal relationship was poor on maximum occlusion (). The cone-beam computed tomography (CBCT) scans showed that the lesion was an amorphous multilocular lytic lesion that had a thinned, well-defined, and scalloped cortical border in some parts (). The enhanced computed tomography (CT) scans revealed a non-enhanced multilocular lesion, which had CT values from -10 to 8 Hounsfield units (HU) (). Magnetic resonance imaging (MRI) showed homogeneous intermediate signal intensity (SI) on T1-weighted images and homogeneous high SI on T2-weighted images, with a clear border in both images. The articular disc of the TMJ was located in a normal position (). A tentative radiographic diagnosis of the simple bone cysts was made, and central giant cell granulomas and aneurysmal bone cysts were included in the differential diagnosis of the lesion.
Surgery was performed under general anesthesia through an incision of the left posterior vestibule. The half of the mandibular ramus containing the mandibular condyle was rejected and eviscerated. The condylar defect with a partially thin cortical plate was revealed. The cortical plate was removed in parts, and the internal walls of the cavities were lightly curetted (). Small amounts of tissue obtained from the defect for microscopic examination were negative for epithelium. The eviscerated bone segment was repositioned to its original state and fixed with miniplates.
Microscopic examination of the specimen showed a cystic lesion without an epithelial lining, which was surrounded by dense fibrous connective tissue. Osteoid bone was also seen in the connective tissue indicating reactive bone formation. These features were consistent with those of simple bone cysts ().
A 14-month postoperative radiographic follow-up showed a recurred lesion at the left mandibular condyle. The lesion had recurred on the previously scalloped part. It showed radiolucency with an expanded, scalloped cortical plate on the CBCT images. A second surgical excision was performed using the preauricular approach. The microscopic features were similar to those of the initial biopsy specimen.
The 6-month postoperative recovery was unsatisfactory even though there was no sign of recurrence. The patient was treated for hemihypesthesia of the left lower face due to a left facial nerve injury followed by a pre-auricular surgical approach.


A 32-year-old man visited our institution for the evaluation of horizontal diplopia, dizziness and headache after the onset of a 5-minute generalized tonic-clonic seizure. On admission, the patient was alert and did not have any possible vascular risk factors such as hypertension, diabetes mellitus, hyperlipidemia, advanced age and cigarette smoking. The attending neurosurgeon and opthalomologist did not find any abnormality except slight nuchal rigidity and bilateral abducens nerve palsy (). Initial cranial computed tomography (CT) () revealed no definite high density lesion in the brain including parenchyma, ventricle and cisternal space, but CT angiography () showed an aneurysm of the ACoA. Magnetic resonance imaging (MRI) () demonstrated focal intraventricular hemorrhage of the posterior horn of the lateral ventricle without abnormalities of the brain stem including infarction or space-occupying lesion. Catheter angiography was followed and a bilobulated saccular aneurysm (6.3×4.0 mm, dome×neck) without compression of brainstem was observed (). Endovascular coil embolization was conducted to obliterate the aneurysmal sac without delay. Continuous lumbar drainage was performed for 6 days. A photograph taken 12 days later illustrated the improvement in both abducens nerve palsy (). The signs of diplopia were resolved completely 9 weeks later (). There were no clinical and radiologic signs of vasospasm during hospitalization.


A 20-year-old-male presented to the emergency department with a history of penetrating injury to the penis by a needle on the same day. The needle was reported to have accidentally pierced the external genitalia at the penoscrotal junction when the penis was erect. There was no history of hematuria. Upon physical examination, the patient was anxious and his vital signs were normal. The caudal end of the needle was just palpable at the penoscrotal junction on local examination.
The results of urinalysis and complete blood examination reports were normal. An X-ray of the pelvis () showed the needle within the pelvis. Ultrasound examination of the abdomen including the genitalia showed a 6-cm needle piercing the prostate from the root of the penis up to the bladder neck. Because the needle was palpable at the penoscrotal junction and the caudal end of the needle was below the urogenital diaphragm (), an urgent surgical exploration with incision over the palpable end of the needle under fluoroscopic guidance was done. This exploration showed the tip of the caudal end of the needle to be inside the corpus cavernosum at the root of the penis (). The needle was manipulated by holding the caudal end at the penoscrotal junction, which showed that the tip of the needle was in and around the bladder neck by piercing the prostate gland. Endoscopic manipulation was not done in this case. The needle was removed intact through exploration of the bulbous cavernosa (). A pressure dressing was applied to the penis and the patients post operative voiding was normal. When the patient was queried about the possibility of self-insertion because accidental injury would be unlikely to push the needle completely through the external genitalia, the patient was too ashamed to admit the cause and was discharged after 2 days. Later, the patient admitted the motive behind the insertion of the needle as sexual gratification. The surgical wound healed well and mild penile edema was noted in the immediate postoperative period. The edema was reduced by a pressure dressing and no complications such as erectile dysfunction or plaque formation occurred during the 1-year follow up after needle removal.


A 53-year-old female was diagnosed with a pancreatic tumor when she initially presented with anorexia and weight loss. Contrast-enhanced CT (CECT) examination at that time revealed a large, homogenous enhancing mass involving almost the entire pancreas with no evidence of local invasion or nodal involvement. A biopsy examination of pancreatic tissue revealed features of a well-differentiated tumor of neuroendocrine origin (neuroendocrine carcinoma proliferating index, 4%). In view of extensive disease involving pancreas, surgery was deferred and the patient was put on octreotide therapy. The patient was apparently well for a period of 4 months when she started having abdominal pains, which progressed in severity over the next month. A repeat contrast-enhanced examination of the abdomen revealed no significant alterations of the primary pancreatic mass, but showed numerous filling defects within the mesenteric vasculature. In view of the disease progression, a staging PET/CT using 68Ga-DOTANOC was performed []. Intense radiotracer uptake was noted in the primary tumor (SUVmax-53). Also seen was an arborizing area of abnormal tracer uptake within the abdomen, which was subsequently traced to mesenteric vessels []. Thus, 68Ga-DOTANOC PET/CT helped confirm the presence of mesenteric vascular thrombosis and localize the cause of abdominal pain to be the same. In addition, the whole body examination revealed no other areas of abnormal uptake that could suggest presence of other metastatic sites.


A 75 year old male patient visited the pain clinic at our hospital complaining of stabbing pain and grouped erythematous vesicles in the external auricle and the C2 dermatome of the ipsilateral temporal region, which started approximately 10 days before (). At the initial occurrence of the symptoms, the patient had been diagnosed with an acute herpes zoster infection at the local ENT and dermatology hospital, so was taking oral antiviral agents for more than a week with no improvements in the symptoms. According to his premedical history, the patient had been undergoing treatment for diabetes mellitus and angina, while taking aspirin, 100 mg/day, and cilostazol, 200 mg/day, (pletaal@). At the time of the hospital visit, the visual analogue scale was 7/10, so the patient was hospitalized. Aspirin and cilostazol were temporarily discontinued and oral drug treatment was started with pregabalin, 150 mg/day, nortriptyline, 10 mg/day, and acetaminophen, 750 mg/day. For the relief of sympathetic mediated pain, a stellate ganglion block was performed daily using 0.375% ropivacaine 6 cc with a paratracheal approach technique on the left C6 level. According to the routine blood tests taken at the time of hospitalization, there were no abnormal observations other than an erythrocyte sedimentation rate (ESR) of 39 mm/hr and a high sensitive C-reactive protein (hs-CRP) of 5.81 mg/dl.
On the morning of the second day of hospitalization, skin eruptions began to appear in numerous areas along the bilateral scalp, posterior neck, shoulder, upper arm, and post upper back (, ). The patient complained of an itching sensation, but no pain in the newly blistered areas. At the same time, there were symptoms of left facial palsy, hearing impairment, otalgia, and tinnitus, and the patient was unable to walk independently due to vertigo with accompanying mild cognitive disability (). His vital signs showed that his blood pressure, heart rate, and respiration rate were normal, but the temperature had risen to a maximum of 38.0℃, while there were no headaches, nausea, or Kernig's signs. Hence, a disseminated herpes zoster infection with accompanying Ramsay Hunt syndrome in the left facial area was suspected, so a blood culture, urine culture, tumor marker (CEA, CA 19-9, PSA), serum test for anti-HIV antibody (ELISA) and anti-VZV IgM antibody (ELISA), and chest x-ray were performed first to differentiate the cause of infection and immune suppression. Immediately after the above symptoms were observed, nephrologists, neurologists, dermatologists, and otolaryngologists were consulted, with the resulting diagnosis being a disseminated herpes zoster infection. In concern for renal injury from old age, the BUN and creatinine figures were observed every 2 days, and acyclovir, 1,500 mg/day, was IV administered for a week. Pregabalin was stopped, as it may have caused the cognitive disability and vertigo, and was replaced with neurontin, 900 mg/day. The stellate ganglion block was continued. Considering the possibility of cerebrovascular disease or myelopathy, a brain stroke MRI was taken on the 3rd day of hospitalization, followed by a T2 sagittal whole body spine MRI on the 5th day of hospitalization, but no abnormalities were observed. The possibility of meningitis from the virus was also suspected, so a CSF culture (body fluid examination, gram stain, fungus, acid fast bacilli smear) was performed, but again no abnormalities were observed. The administration of steroids was considered for the Ramsay Hunt syndrome, but it was not attempted due to concerns of immune suppression. No abnormalities were observed in the additionally performed cultures (blood, urine, CSF), tumor markers, anti-HIV antibodies, and anti-VZV IgM antibody tests. The hs-CRP was high at 2.44 mg/dl, but it was lower than at the time of hospitalization, so there was a gradually decreasing trend that recovered to normal figures before discharge from the hospital. While maintaining drug therapy and observing the progress, all vital signs returned to normal on the 4th day and the skin lesions did not develop further after the 6th day of hospitalization. Gradually, the vertigo and tinnitus symptoms also improved, so by the 10th day the patient recovered enough to walk independently. The patient began to recover from the facial palsy and hearing impairment from the 9th day, so the patient was discharged according to his wishes on the 12th day while maintaining outpatient treatment at the clinic. The patient visited the clinic every week for two months after his discharge, and there were nearly no complications.


A 57-year-old man visited Seoul National University Hospital due to an incidentally detected hilar mass. It was a 4.2 cm mass encasing the left main pulmonary artery and involved the left main bronchus within 2 cm from the carina but not the carina itself (). Bronchoscopic examination showed an exophyting mass in the left upper lobar bronchus and tumor infiltration in the left main bronchus. A hypermetabolic mass in the left hilum was identified without metastasis by a positron emission tomography-computed tomography scan. He underwent left intrapericardial pneumonectomy and mediastinal lymph node dissection by hemi-clamshell incision after mediastinoscopic lymph node biopsy in April 2007. The pericardium was resected in en bloc fashion and the pulmonary artery was divided in the pericardial cavity. The pericardial defect was reconstructed with a Gore-Tex patch. The patient was discharged on postoperative day 15 without any complications. The final pathologic result reported a clear resection margin at the bronchial and pulmonary arterial stump; however, there was microscopic single nodal metastasis at the subcarinal lymph node.
On regular outpatient follow-up, the patient was readmitted on diagnosis of postoperative empyema with bronchopleural fistula (BPF) and received a window operation in March 2008 (). However, for fear of exposure of the pericardial cavity externally, we did not remove the Gore-Tex patch at that time. The BPF closed spontaneously several months after the window operation; however, the remaining Gore-Tex patch caused repeated infection and continuous dirty discharge. Therefore, we removed the Gore-Tex patch in October 2009 and noted that the heart was covered with a thick peel of empyema.
Six months after removal of the Gore-Tex patch, we screened for any cancer recurrence and decided to perform a Clagett procedure. As a first step, a widening of the window was performed to create enough space for the irrigation and debridement to follow. A vacuum-assisted closure (VAC) device was applied at postoperative day 1, and VAC sponge change was repeated at postoperative day 6, 9, and 12 (). Finally, the Clagett procedure completed by filling the pleural space with antibiotic solution at postoperative day 14 and the patient was discharged without complications ().
For a 1-year period of outpatient follow-up, the cavity was fully filled with pleural effusion, and there was no recurrence of empyema or lung cancer.


A 25-year-old female presented with a painless lump in her right breast for 1 month duration. She gave history of low grade fever off and on for the last 2 weeks. There were no other complaints like weight loss, loss of appetite, and any cough. She was unmarried and there was no positive family history of breast tuberculosis. On local examination, a non-tender lump of size 3 × 4 cm was felt in lower quadrant of the breast [marked with a line as a - ]. It was freely mobile and no axillary nodes were present. Another 1 × 1.5 cm swelling was present in infra-mammary area just below and medial to the above mentioned lump [marked by line b; ]. Her systemic examination was non-contributory.
On routine blood investigations, total leucocyte count was 12,000 mm3 and erythrocyte sedimentation rate (ESR) was 60 mm at the end of 1 h, and HIV test was negative. Her fasting blood sugar and blood urea levels were normal. Chest X-ray was normal. Fine needle aspiration cytology (FNAC) of the breast and infra-mammary lump showed granulomas, epitheloid cells, and mixed inflammatory cells []. Background consisted of necrotic material and ZN stain for acid fast bacilli (AFB) was positive in the FNAC of both lumps. Mountex test was not done as on FNAC diagnosis already was made. Final diagnosis made as breast TB.
She was put on a 6-month course of anti-tubercular therapy with a 2 month intensive phase of rifampicin, isoniazid, ethambutol, and pyrazinamide followed by a consolidation phase of rifampicin and isoniazid for another 4 months. The lump size was decreased within 3 weeks of initiation of ATT and at 3 month follow-up the patient remains asymptomatic and both lumps disappeared. She was advised to continue treatment.


An 8 months old female infant weighing 6.5 kg brought by her mother to the plastic surgery clinic of the University of Benin Teaching Hospital with complaint of her mouth being wider than normal since birth. A third child in a monogamous setting from Effurun, Delta State, Nigeria. No family history of similar condition. Her mother never smoked nor took alcohol neither was she on any known anticonvulsants; but ingested postinor several times while pregnant because pregnancy was unwanted for financial reasons. No other means of termination was attempted. Although there was no antenatal care, she was delivered by spontaneous vaginal delivery, cried immediately after birth and had normal developmental milestones. Family's source of drinking water was from an untreated borehole. She had no difficulty with feeding and was exclusively breastfed for 2 months.
Close examination revealed a cheerful infant with bilateral transverse facial cleft, Tessiers no 7 also referred to as congenital macrostomia or commissural cleft [] which was more evident either on smiling or crying []. Thorough examination and investigation revealed neither associated congenital anomaly nor any coexisting medical illness. Repair by bilateral Z plasty was done by the plastic and burns unit.


A 13-year-old-boy reported to the Department of Pedodontics and Preventive Dentistry with the chief complaint of swelling and pain in the lower left back teeth region. On clinical examination, extra oral swelling which was tender and hard in consistency extending from the anterior border of the left ramus to the left parasymphysis region measuring approximately 4×3 cm [] was observed. On physical examination, presence of dysmorphic facial features like relative macrocephaly and ocular hypertolorism were observed. Sprangal scapular deformity [] and palmar and planter pits [] were also observed. On intraoral examination, grossly decayed mandibular left second primary molar and swelling in the vestibule region was observed.
Intraoral periapical radiograph revealed the presence of large radiolucent area with sclerotic border, suggestive of a cyst. Orthopantomograph revealed the presence of multiple cysts both in maxilla and mandible []. Considering the possibility of the NBCCS with the above features, further investigations were carried out. Computed tomography showed ectopic calcification of the falx cerebrai [] and presence of multiple cysts in the maxilla [] and mandible []. Antero-posterior view of the chest showed the presence of bifid rib in the posterior aspect of the right 3rd rib and rib expansion was noted in the anterior end of the left 3rd and 4th rib []. Lateral cephalograph showed bridging of the sella turcica [].


The patient presented in this case report was a 63-year-old male with a previous history of papillary renal cell carcinoma (RCC) of Fuhrman Grade of II for which he received a left radical nephectomy at age 51 in an outside facility. He presented with a 4.33-cm lower pole tumor of his solitary right kidney diagnosed via contrast-enhanced computed tomography (CT) scan [] and came to our facility for evaluation. In addition to previous RCC, this patient had an age-adjusted Charlson Comorbidity Index of 5 and a preoperative creatinine of 1.01 with an estimated glomerular filtration rate (eGFR) of 79 ml/min using the modification of diet in renal disease (MDRD) calculation. Treatment options including tumor extirpation and surveillance were offered as alternatives, and the risk and benefits of MWA were discussed with the patient.
A transperitoneal approach was used. After exposure of the kidney and localization of the tumor, two 13-gauge, 3.7-cm MWA surgical antennae (Covidien, Valleylab, Boulder, CO, USA) were introduced through separate incisions. Fiber-optic thermal sensors were placed at the tumor periphery via visual and ultrasound (US) guidance utilizing a coaxial guide needle with radiopaque sheath (Huey, Cook Vascular, Inc., Vadergrift, PA, USA). Prior to ablation with the antenna(e) in place, a spring-loaded 16-gauge biopsy needle was utilized to take multiple biopsies under visual and US guidance. The microwave antenna was advanced into the tumor [] and MWA commenced with simultaneous peripheral fiber-optic thermometry (Lumasense, Santa Clara, CA, USA) to determine treatment endpoints of >60°C. The microwave system used was the ValleyLab Evident® 915 MHz MW ablation system (Covidien Inc., Boulder, CO, USA) with two 13-gauge, 3.7-cm tip antenna(e) driven at 45 W. Ablation was performed for 23 min at 45 W over three separate deployments. This was based on when the tumor periphery temperatures reached the target temperature as in the techniques previously published.[] Tumor biopsy showed papillary type RCC, Fuhrman grade II.
The patient had an uneventful postoperative course, and was followed with a contrast-enhanced CT scan at 1 month [] and at 4 months []. The scan performed at 4 months post-op showed nodular thickening with enhancement at the ablation site read as recurrence [].
A transperitoneal approach was used. The procedure was performed using the same method as described above. Three 3.0-cm Cooltip® RFA probes (Covidien Inc.) were positioned in a triangular array with peripheral thermometry as described above []. Ablation was performed for a total of 31 min with all peripheral thermometry exceeding 60°C. Tumor biopsy at that time showed unspecified RCC. The patient had an uneventful postoperative course and was followed with a 1-month, 11-month, and 24-month clinical follow-up with 4-phase contrast-enhanced CT scan showing no enhancement of the tumor ablation site and no evidence of recurrent disease. Creatinine values pre-MWA, post-MWA, and post-RFA were 1.01, 1.14, 1.17 mg/ml, respectively. This represents a change in eGFR from 79 to 67 ml/min post-MWA (decreased 15%), and no change post-RFA.


A 20-year-old female presented to us with slowly growing breast lump for last 1 year, localized in upper outer quadrant. The lump was well defined, freely mobile and measured 3 × 3 cm in greatest dimension. The overlying skin was unremarkable. Axillary palpation was negative for lymph nodes.
Fine needle aspiration was performed. The cytological findings revealed cell rich smears comprising monomorphic loosely cohesive sheets of plasmacytoid cells, round to polygonal cells with round to ovoid eccentrically placed nuclei, finely dispersed chromatin, and moderate amount of cytoplasm. Overall findings suggested myoepithelioma, breast – plasmacytoid type [].
On the basis of above findings lumpectomy was done and the tissue was sent for histopathological examination. Grossly, the tissue was well circumscribed greyish white measuring 3 × 3 cm. Microscopic examination revealed tumor cells arranged in sheets with abundant myxoid stroma. The individual cells were round to polygonal, having prominent eosinophillic cytoplasm and eccentrically placed round nuclei. The diagnosis of myoepithelioma, plasmacytoid type was made on the basis of the above findings.
On immunohistochemistry cells were positive for S-100 protein and actin.


A previously healthy 19-year-old man admitted with a 2-day history of headache. The headache was localized to the occipital area, severe in intensity and throbbing in nature. Typically, the headache in this patient was position-dependent, and developed with orthostatic posture, leading to severe intensity in the upright position (visual analogue scale [VAS] 9–10/10), and was relieved when the patient was in the recumbent position. Routine neurologic examination and laboratory findings were normal. Initial computerized tomography (CT) of the brain showed no abnormalities. Cerebrospinal fluid (CSF) tapping was done under the suspicion of SIH, but the opening pressure was over 40 cmH2O (normal range: 8-18 cmH2O), indicating increased intracranial pressure (IICP). After admission, the patient developed left upper extremity monoparesis with intermittent clonic movement, followed by generalized tonic movement. Diffusion-weighted magnetic resonance imaging (MRI) (DWI) of the brain was performed immediately after the episode. The b-value of 1000 s/mm2 and apparent diffusion coefficient (ADC) image of the DWI showed high signal intensity in the right parietal cortex, suggesting vasogenic edema [Figure -]. Gradient-echo imaging (GRE) identified superior sagittal sinus (SSS) and cortical vein dilatation [Figure and ]. Subsequent MRI revealed right parietal, left frontal cortex high-signal intensity on T2-weighted and fluid-attenuated inversion recovery images, with an empty delta sign on gadolinium-enhanced T1-weighted images [Figure -]. There was no evidence to suggest SIH on MRI, such as pachymeningeal enhancement or downward brain displacement. Brain MR venography (MRV) revealed the absence of the SSS, straight sinus and right transverse sinus [Figure and ]. We made a diagnosis of CVT with venous infarction according to the established criteria[] to which the patient's headache and seizure were attributed. Further laboratory tests for causative medical conditions showed deficiencies in protein C (16%, normal range: 70- 130%) and protein S (33%, normal range: 74- 146%). The headache and seizure completely subsided after intravenous anticoagulation treatment. He remained asymptomatic without any neurological sequelae after 10 months of follow-up.


A previously healthy 29-year-old man visited the hospital complaining of headache for the past day. The headache was localized to the bifrontal–temporal area with moderate to severe intensity (VAS 7–8/10), and was squeezing in nature. The symptoms developed only when the patient stood up, and were partially relieved when lying down (VAS 2-3/10). We performed CSF tapping and checked intracranial pressure under suspicion of SIH. However, the CSF opening pressure was 36 cmH2O (normal range: 8-18 cmH2O). MRI of the brain showed no definite parenchymal lesion, but gadolinium-enhanced T1-weighted images revealed the empty delta sign with enhancement of the collateral veins in the SSS walls surrounding a non-enhanced thrombus in the sinus. Also, there were no findings suggesting SIH. MRV revealed a voiding defect in the SSS, right transverse sinus and right sigmoid sinus. Diagnosis of CVT according to the clinical presentation and imaging findings was made. The patient's headache near completely subsided after intravenous anticoagulation treatment. He remained asymptomatic after 13 months of follow-up.


A 40-year-old man with status epilepticus, residing in Kolkata, was admitted to our hospital. Two years before his admission, he had experienced the first episode of generalized seizure, for which he had been receiving sodium valproate. He had been experiencing recurrent episodes of generalized tonic clonic seizures for 2 days before his admission despite the continuation of sodium valproate in regular doses. He had a history of progressive hearing loss for the last 2 years and perioral paresthesia for the last 2 months. He did not have a family history of similar illness, head trauma in the past, or fever. Neuroimaging or electroencephalography (EEG) was not performed previously. His vital parameters were within the normal range, except that he had tachycardia. Neurological examination showed depression of deep tendon reflexes in both upper and lower extremities. He exhibited no neurological signs, except for Chvostek's and Trousseau's signs.
Non-contrast computed tomography (CT) of the brain showed extensive intracranial calcification involving the basal ganglia, cerebellum, and cerebral cortex on both the right and left sides []. Electrocardiography during the immediate postictal phase showed a prolonged QT interval. The results of routine blood examination were as follows: hemoglobin, 14.7 gm/ dL; (14-16 gm/dL) erythrocyte sedimentation rate, 20 mm during the first hour; <5 mm 1st hour random blood glucose, 142 mg/dL; albumin, 4.5 g/ dL; (3.5-5.5 gm/ dL) urea, 26.0 mg/dL (normal range, 10–20 mg/dL); creatinine, 2.2 mg/ dL (normal range, 0.6–1.2 mg/dL, estimated glomerular filtration rate - eGFR ~32 ml/min)); sodium, 146 mmol/dL (normal range, 137–145 mg/dL); potassium, 3.6 mmol/ dL (normal range, 3.5–5.5 mg/dL); calcium, 3.8 mg/ dL (normal range, 8.8–10.6 mg/ dL); phosphorus, 9.78 mg/dL (normal range, 2.7–4.5 mg/dL); and magnesium, 0.7 meq/L (normal range, 1.3–2.5 meq/L). Hormonal assay showed that the level of 1,25-dihydroxyvitamin D3 was 10.9 pg/mL (normal range, 19.6–54.3 pg/mL); electrochemiluminescence immunoassay (ECLIA) showed that the serum intact parathyroid hormone (PTH) level was 1.44 pg/mL (normal range, 15–65 pg/mL). The patient did not have any history of neck surgery or radiation exposure that could have led to hypoparathyroidism.
Pure tone audiometry (PTA) showed bilateral moderate-to-severe hearing loss that was more severe at the higher end of the frequency spectrum [Figure and ]. Ultrasonography showed the absence of renal shadow on the left kidney and an increased cortical echogenicity of 8.4 cm in the right kidney []. Because of the simultaneous occurrence of hypoparathyroidism, deafness, and renal agenesis, the patient was diagnosed with HDR syndrome. However, chromosomal analysis with trypsin G-banding yielded normal results.
The patient was administered intravenous phenytoin, levetiracetam, and calcium gluconate. After the seizures were controlled, he was administered oral phenytoin, levetiracetam, calcium carbonate, and alfacalcidol. He was discharged on the 10th day and is being followed up.


A 55-years-old male weighing 60 kg was diagnosed with bilateral vitreous hemorrhage and scheduled for pars plana vitrectomy. He was known hypertensive since 1 year and was managed with oral amlodipine (5 mg) twice a day and losartan (50 mg) once a day. Since 4 months, patient started having progressively increasing loss of vision and memory loss which was followed by sudden onset headache associated with vomiting and subsequently loss of consciousness. Pupils of left eye were dilated and sluggishly reacting while right eye was of normal size and sluggishly reacting. Magnetic resonance imaging (MRI) of head revealed left frontal intracranial hemorrhage with intraventricular extension. Diagnosis of stroke with left frontal bleed was made. He was kept on conservative management with continuation of antihypertensive drugs. The patient's consciousness gradually improved. GCS was E4 M6 V5, motor power was 5/5 with down going planters. He had a full recovery after 1 month. The vision did not improved and there was progressively increasing loss of vision. On further evaluation, it revealed hypertensive bleed in the eye and magnetic resonance angiography was done which could not rule out aneurysmal bleed. He was planned for pars plana viterectomy.
Preoperatively, patient was conscious and maintaining vitals. His routine investigations including 12 lead electrocardiogram, chest X-ray, hemogram, liver, and kidney function test were normal. His antihypertensive drugs were continued and was premedicated with oral alprazolam (0.5 mg) night before the surgery. He was also administered oral ranitidine (150 mg) night before the surgery and in the morning after the surgery. A fasting status of 6 h for solid food and 2 h of clear fluid was advised.
In the operating room, monitors including 5 lead electrocardiogram, non invasive automated blood pressure, and pulse oximeter were attached. After securing intravenous access, intravenous midazolam (1 mg) and fentanyl (120 μg) were administered. Anesthesia was induced with propofol (120 mg) and lungs were ventilated with 3% sevoflurane in nitrous oxide and oxygen (50:50). After achieving neuromuscular blockade with rocuronium (40 mg) and intravenous administration of preservative lidocaine (60 mg), airway was secured with proseal laryngeal mask airway (PLMA). Anesthesia was maintained with sevoflurane in nitrous oxide and oxygen (MAC 1) and boluses of rocuronium (10 mg) and fentanyl (20 μg). After the surgery, the residual neuromuscular blockade was reversed with neostigmine (2.5 mg), glycopyrollate (0.4 mg) and PLMA was removed after adequate respiratory efforts were present. The patient had an uneventful recovery and discharged 3 days later for further follow up in ophthalmology and neurology clinics.


A 35-year-old woman with the following characteristics was posted for emergency caesarean delivery in view of intra-uterine growth retardation and foetal tachycardia: Second gravida at 35 weeks of gestation with TA, AR and hypertension for 5 years, on treatment with tablet methyldopa 500 mg, amlodipine 5 mg and extended release metoprolol 50 mg, each, twice daily.
Her pulse rate was 114/min and BP was 183/78 mmHg in right arm supine position. Volume of upper limb pulses appeared to be bilaterally equal on palpation. However, right radial pulse was collapsing in nature along with radio-femoral delay. Grade four early diastolic murmur, loudest in aortic area, with palpable thrill-radiating to left sternal border was present. Examination of other systems and investigations were unremarkable. Two dimensional (2D) echocardiogram revealed narrowing of thoracic and abdominal aorta to 9 mm with a high blood flow velocity of 2.0 m/s. Ascending aorta and aortic arch were dilated along with moderate AR. Left ventricular ejection fraction was 58% with normal biventricular systolic function and normal pulmonary artery pressure. Doppler performed 5 years back showed high resistant flows in common, internal and external carotid arteries bilaterally, with possible proximal stenosis of right subclavian artery (SCA).
Baseline vitals in the operating room were as follows: Heart rate of 115/min, non-invasive blood pressure (NIBP) of 188/78 mmHg in right upper limb and SPO2 of 97% [].
Anaesthetic plan was lumbar epidural anaesthesia with invasive radial arterial BP monitoring prior to placing the epidural. Since the doppler report showed possible stenosis of right SCA, left radial arterial line was secured and the invasive BP (IBP) was 112/55 mmHg. Lower limb arterial monitoring was not considered due to narrowing of thoraco-abdominal aorta. Epidural catheter was secured in L1-L2 intervertebral space, and the position was confirmed with test dose of 3 ml of 2% preservative free lignocaine. Patient was positioned supine with a wedge under the right buttock. To achieve a sensory block up to T6 level, 10 ml of 0.5% ropivacaine was given in 5-ml increments every 10 minutes. There was a drop in IBP to 80/40 mmHg along with a drop of NIBP to 122/60 mmHg after the epidural over 10 minutes. We treated the drop in IBP with 6 mg ephedrine boluses to avoid any cerebral or utero-placental hypoperfusion. There was ill-sustained increase in IBP and NIBP after each bolus (3 boluses provided). BP increased up to 122/64 mmHg, while the NIBP increased up to 198/94 mmHg after the third bolus of ephedrine. Patient complained of headache after each bolus of ephedrine. Possibility of over treating IBP leading to headache was considered after ruling out possibilities of monitoring error. On-table doppler ultrasound of upper limb arteries was performed specifically looking at the luminal diameter and amplitude of pulsations. Both were reduced in the left radial and axillary artery when compared to the right radial and axillary artery. Right radial pulsation was collapsing in nature, which is expected in AR. Thereafter, we considered treating using the NIBP values of the right upper limb. Meanwhile, the baby delivered was shifted to neonatal intensive care unit in view of prematurity. Rest of the intra-operative period was uneventful. Postoperatively, the patient was monitored in a high dependency unit. Post-operative aortogram revealed normal right SCA and diffuse disease of the left distal SCA, which confirmed our intra-operative findings.


A 16-year-old female presented with a 3-month history of progressive upper back pain. She showed a visual analogue scale result of 50 mm, with the pain being worse in the supine position at night. On the first visit, she was treated with a conservative management. However, the pain did not subside and a sudden weakness of the lower extremities developed after 5 days. She reported a radicular pain along the T1 dermatome and hypoesthesia below the T1 dermatome, along with constipation. This patient's neurologic classification according to the American Spinal Injury Association (ASIA) International Standards for the neurological classification of the Spinal Cord Injury (SCI) is as follow: sensory and motor level of injury, T1 bilaterally; and degree of completeness, ASIA Impairment Scale (AIS) grade D. She demonstrated 3+reflexes at the both patellas and ankles and a positive Babinski sign. The Modified Ashworth Scale (MAS) was graded as 1. Her strength was generally of a fair grade in the lower extremities. There was tenderness on percussion of the T2 thoracic spine. On the radiographs, there was an osteolytic bony lesion at the T2 vertebra. The magnetic resonance imaging (MRI) revealed a large mass in the T2 vertebral body with the involvement of the left pedicle, transverse process, both lamina and spinous process. In addition, there was high signal intensity in the spinal cord at the T2 level, which suggested compressive myelopathy ().
After the patient was transferred to the department of neurosurgery, she was treated with a tumor resection and laminectomy of the T2 vertebra, with the posterior fixation of T1, T3, and T4 (). On the pathologic report, the large mass was confirmed to be GCT (). To optimize the local disease control, the patient underwent the CyberKnife (Accuracy Inc., Sunnyvale, CA, USA) three times over three days. The postoperative anterior-posterior plain film and the lateral plain films of the thoracic spine revealed a good screw position ().
Six weeks after the surgery, the patient was transferred to the department of physical medicine and rehabilitation. The radicular pain was subsided, but the mild hypoesthesia from T1 to T3 dermatome remained. She demonstrated no hyperreflexia and fair+ to good strength in the lower extremities. The MAS was graded as 0. A moderate assistance was required for walking and the lower body dressing. The patient was placed in a cervicothoracic orthosis for stabilizing the operative site. She practiced walking with a high walker and a cane later. She was trained in using a dressing stick. The function of the bladder was normal when checked with a bladder chart and the measurement of the residual urine. However, the patient complained of constipation. She was given a high-fiber diet regimen, along with the timed bowel program and the abdominal wall massage. As a result, her constipation subsided. After one month, the patient was able to walk independently, and the impairment scale was changed to AIS E (sensory and motor functions are normal) without any evidence of recurrence (). The patient is now two years out of the surgery and the radiation therapy. She has returned to school as a student, has no significant complaints and remains neurologically intact.


A 65-year-old male was admitted for abdominal pain and fever, which started 2 weeks ago. He was previously healthy without any specific past medical history. Abdominal physical examination revealed tenderness without rebound tenderness on epigastrium. Laboratory finding demonstrated white blood cells 12.3×103/µL, hemoglobin 11.6 g/dL, platelets 309×103/µL, total bilirubin 0.7 mg/dL, alkaline phosphatase 117 IU/L, aspartate aminotransferase 26 IU/L, alanine aminotransferase 33 IU/L, high sensitivity C-reactive protein 22.1 mg/dL, carcinoembryonic antigen 2.0 ng/mL, carbohydrate antigen 19-9 7.6 U/mL, and alphafetoprotein 1.3 ng/mL. Hepatitis B surface antigen was negative and hepatitis C virus antibody was positive. Abdominal computed tomography (CT) showed about 9 cm lobulating low attenuating mass with peripheral rim enhancement in the left lobe of the liver, which was thought to be mature pyogenic liver abscess (). He started antibiotics therapy with ceftriaxone and metronidazole, and underwent percutaneous drainage tube insertion using 10.2 Fr pigtail catheter into the liver abscess. Two days later fever subsided and a week later he was discharged with oral antibiotics. One month later, follow-up liver CT scan showed 1.5 cm enhancing lesion in distal CBD with progression of biliary dilatation, suggesting distal CBD cancer (). Thus biopsy was performed at CBD via endoscopic retrograde cholangiopancreatography (), which revealed adenocarcinoma (). Additional CT scan () and magnetic resonance imaging (MRI) () showed multiple small nodules in right hepatic lobe and metastatic adenocarcinoma was confirmed from frozen slide of sono-guided liver biopsy (). Since the patient was inoperable, he underwent combination chemotherapy with gemcitabine 1,600 mg intravenous infusion on day 1 and S-1 (tegafur+gimeracil+oteracil) 500 mg twice a day for 14 days with 1-week rest as one course. Serial CT scans checked every three cycles showed gradual regression of the metastatic nodules in liver (). After nine courses of chemotherapy the hepatic lesion completely disappeared on MRI (). Three weeks later, pylorus preserving pancreaticoduodenectomy was performed. Grossly the resected CBD showed a 1×1 cm sized gray solid mass infiltrating into adjacent soft tissue. Microscopically there were no residual tumor but chronic active cholangitis with multifocal erosion and subepithelial fibrosis in the resected CBD (). Then, 22 days after the operation he was discharged without any complication. For 3 months of follow-up, he did not develop any signs of recurrence and no evidence of recurrence was found in the 3-month postoperative CT scan ().


A 37-year-old man with keratoconus () could not tolerate RGP lens-wearing and did not want to have a corneal transplant or other corneal surgeries. He had been using RGP contact lenses in both eyes for 10 years. He was diagnosed with unilateral keratoconus of the left eye. Stable keratoconus was confirmed with refraction and corneal topography for 7 years (). In the right eye, the UCVA was 0.04 and the BSCVA was 0.7 with -3.75 -3.00 × 155A. In the left eye, the UCVA was 0.02, and the BSCVA was 0.4 with -12.00 -3.50 × 30A. The corneal refractive power was 58.40 / 48.70 diopters × 117A in the left eye (). With manifest refraction, a BSCVA of 0.4 was achieved. He was told that the maximal possible visual correction would be 0.4 or less because irregular astigmatism could not be corrected with toric ICL implantation. With informed consent, toric ICL implantation was performed without any surgical complications. After surgery, the manifest refraction became -0.75 × 180A with a UCVA of 0.5 and BSCVA of 0.7 at 1 month, which then shifted to +2.25 -0.50 × 180A with a UCVA of 0.4 and BSCVA of 0.7 at 9 months, and to -1.75 × 180A with a UCVA of 0.2 and BSCVA of 0.5 at 20 months. The anterior chamber depth, from the endothelium to the ICL, decreased from 2.21 mm at 1 month to 2.13 mm at 9 months follow-up, corresponding with mild corneal flattening and transient hyperopic changes (), without any vault changes (). At the last follow-up, 20 months after surgery, the ICL vault was decreased, resulting in mild deepening of the ACD from 2.13 mm to 2.17 mm, and the manifest refraction results finally stabilized at mild myopic astigmatism (). The efficacy and safety index were 1.0 and 1.75 at 9 months and 0.5 and 1.25 at 20 months (). Postoperative contrast sensitivity was also enhanced ().


A 9-year-old boy from northern Iran was referred to the Children's Medical Center, the Pediatrics Center of Excellence in Tehran-Iran, for evaluation of high fasting plasma glucose. He was from unrelated parents, and his mild hyperglycemia has been accidentally discovered a few months before during routine laboratory tests. He did not have polydipsia and polyuria, neither any weight loss.
On examination, he was well and healthy, weight and height 43 kg and 148 cm respectively (>97th percentile of Iranian boys of corresponding age) with body mass index (BMI) of 24 kg/m2. He had no acanthosis nigricans. Fasting blood sugar (FBS) 114 mg/dL, HbA1c 6.5%. A standard oral glucose tolerance test (OGTT) with 75g of glucose equivalent was performed with a fasting glucose of 125 mg/dL and 2-hour glucose of 133 mg/dL. Fasting insulin concentration was 7.5 µIU/mL. No glucosuria or ketonuria were detected. Further investigations revealed normal liver function test, lipid profile, and abdominal ultrasound. In addition, the pancreatic islet cell autoantibodies (ICA) and glutamic acid decarboxylase autoantibodies (GADA) were negative. Both clinical presentation and laboratory data were highly suggestive of a GCK mutation.
Family history disclosed a diagnosis of diabetes mellitus in his paternal grandmother 10 years ago, at the age of 55 years, but she took no medication. Because there was no history of diabetes in his parents, evaluation was performed. The mother was healthy, with FBS of 88 mg/dL and blood glucose of 94 mg/dL at 120’ of OGTT. Instead, the 41-year-old father had an FBS of 115 mg/dL and his blood glucose was 162 mg/dL at 2-hours after GTT; his weight was 95 kg, his height 185 cm, with a resulting BMI of 27.9 kg/m2. These findings showed that diabetes or IFG/IGT was present in three consecutive generations in this family and reinforced the clinical diagnosis of MODY, type 2.
Consequently, after getting written informed consent, direct DNA sequencing of all exons and intron-exon bounders of the GCK gene was performed on patient's genomic lymphocytes DNA, which showed a heterozygous single nucleotide deletion in exon 8 (c.1010delA) causing a frame-shift with the formation of a premature stop codon (TGA) downstream at codon 352 (exon 9) (). The same mutation was found in his father as well. Therefore, the diagnosis of MODY2 was confirmed in the family.
Consequently, after getting written informed consent, direct DNA sequencing of all exons and intron-exon bounders of the GCK gene was performed on patient's genomic lymphocytes DNA, which showed a heterozygous single nucleotide deletion in exon 8 (c.1010delA) causing a frame-shift with the formation of a premature stop codon (TGA) downstream at codon 352 (exon 9) (). The same mutation was found in his father as well. Therefore, the diagnosis of MODY2 was confirmed in the family.
Serial blood glucose measurements in the patient obtained by glucometer at home showed variable fasting readings ranging between 115 mg/dL and 155 mg/dL; fasting insulin concentration was 6.4 µIU/mL, HbA1c was 6.6%. A second OGTT showed blood glucose of 117, 134, 128, and 118 mg/dL at 0, 60, 120, 180 minutes, respectively, confirming a non-progressive, mild alteration of glucose metabolism. The patient received some dietary advices and no medication and a follow-up appointment in the diabetes clinic. His last biochemical tests after 4 months showed FBS of 127 mg/dL. His latest HbA1c was 6.4%. We recommended further dietary restriction and regular follow-up.


An 18-year old girl reported to the Department of Conservative Dentistry & Endodontics with pain in her right mandibular first molar (#46). On examination a carious exposure was found in the tooth #46. The tooth showed a negative response to electric pulp testing (EPT) and cold testing. The intra-oral periapical (IOPA) radiograph also revealed slight periapical changes in relation to tooth #46. Therefore a diagnosis of pulp necrosis was made and endodontic treatment was started in tooth #46. The access opening was made and the canals were located and the working length was established with size 20 K stainless steelhand files (Dentsply Maillefer, Ballaigues, Switzerland). During the cleaning and shaping procedures for coronal shaping, a ProTaper shaper SX (Dentsply Maillefer) separated in the mesiolingual canal of the tooth.
On taking the radiograph the separated instrument fragment was found within the coronal portion of the canal (). The fragment was well past the orifice of the canal and far enough to be removed by forceps. Removal of the fragment was attempted with various small forceps and pliers but without success, as a sufficient grip of the fragment could not be established. Ultrasonic scaler was also used but the instrument could not be loosened probably because it was locked within the dentinal wall of the root canal. Therefore an alternative method to retrieve the instrument fragment was used as follows:
Gates Glidden drill (GG No.3) was used to remove dentine surrounding the coronal end of the separated fragment (). An 18-guage needle was modified by cutting it with a carborandum disc from the tip to transform it into a microtube (). The modified needle microtube was then inserted into the canal so that it engulfed the coronal end of the separated fragment. Some cyanoacrylate glue was dropped into the microtube and then the tube was placed again into the canal engulfing the coronal end of the separated fragment. The outside of the tube was coated with petroleum jelly. The modified needle microtube was kept in place without disturbance for about three minutes to ensure proper adhesion of the separated fragment with the microtube. The separated fragment adhered to the microtube was then removed as a single unit by giving slight counterclockwise rotation and simultaneous pull out motion (). A confirmatory radiograph was taken and the working length was then reconfirmed (). The cleaning and shaping was completed and the tooth was obturated with laterally condensed gutta-percha and zinc oxide-eugenol based sealer (). The tooth stays in normal function two years after the endodontic treatment ().


A 48-year-old man was referred to our hospital due to a lung mass of unknown origin. The patient had a 60 pack-year smoking history. Prior to admission, the patient visited an outside hospital with a one-month history of dyspnea, cough and sputum. Chest imaging studies demonstrated a lung mass (initial size, 7.7×6.4 cm) at the right mediastinal border with deviation of the trachea. After placing an endobronchial stent in the left main bronchus from the carina, the patient was transferred to our hospital.
Chest X-ray () showed a large mass in the right paratracheal area, causing tracheal deviation to the left with mild bronchopneumonia in the right lower lobe. The stent in the left main bronchus remained patent. Computed tomography (CT) revealed a 7.7×6.4 cm lobulated, heterogeneous enhancing mass abutting the trachea, carina, and both main bronchi, as well as multiple enlarged lymph nodes in the subclavian, right hilar, subcarinal, upper and lower paratracheal areas (). A metallic stent was placed in the left main bronchus and irregular narrowing of the right main bronchial wall was also observed. Flexible bronchoscopy (FBS) showed the placement of a stent from the carina to the left main bronchus, extending to the left second carina (). The left upper and lower bronchi appeared normal. A white fungating mass obstructing the right main bronchus and a pin-point opening due to a mass-like lesion were detected.
Thoracoscopic biopsy of the mediastinal lymph node was performed. Gross examination revealed a single, 2 cm, white, well-defined mass that bled easily. Histopathologically, the mass was composed of numerous glands showing an endometrioid morphology, occasional squamoid morules (×400), and relatively clear cytoplasm (). Based on these histopathological findings, we finalized the diagnosis as fetal adenocarcinoma.
Due to the locally advanced state of the lung cancer in this case, concurrent chemoradiation therapy (Tomo-Helical; 5,500 cGy) was administered. Treatment was comprised of 25 mg/m2 docetaxel weekly for six weeks with concomitant radiation therapy; the patient completed the planned treatment regimen. Repeat CT performed at a 1-month follow-up visit () showed the mass in the right paratracheal area had decreased in size (5.8 cm decreased to 4.6 cm), indicating a stable disease. Additional follow-up chest CT and FBS were scheduled at 3-month intervals. On follow-up studies at 4 months, positron emission tomography-CT showed significantly decreased size and activity (5.5×4.6 cm to 4.5×3.4 cm) of the mediastinal mass, which was considered a favorable response to the treatment, and no other newly developed hypermetabolic foci to suggest distant metastasis were seen. Bronchoscopy () showed relief of the airway obstruction of the right and left main bronchi, with increased ease of passing the scope. Compared with the previous chest X-ray (), the airway obstruction and tracheal deviation were improved (). We were able to remove the stent from the left main bronchus. Previous symptoms of dyspnea and cough were also improved. A further partial response was noted on the 7-month follow-up studies, and another CT follow-up is planned in 3 months.


A 63-year old male from a rural area came to the surgical OPD with a polypoidal mass in the anal region. The mass was gradually increasing in size for past 20 years. He gave the complaint of pain and intense itching over the mass. On examination, the growth was from the anal wall, with skin covered and firmness in consistency measuring about 4 × 3 cm [Figures and ]. All the basic investigations done were within normal limits except a slight increase in the eosinophil count. Stool examination was negative for any worm or its ova. A differential diagnosis of squamous papilloma, fibroma, and foreign body granuloma were considered. Patient was admitted into the hospital and mass from the anal region was excised completely. Lateral sphincterotomy and Balal's stitch was done. Then the specimen was sent to the department of pathology where it was examined and processed.
To our surprise, histopathologic examination of the skin-covered lesion revealed the fragments of dead E. vermicularis worms surrounded by granulomatous reaction composed of epithelioid macrophages, foreign body type of giant cells and lymphocytes [Figures and ]. The granuloma was of the noncaseating type. Sections also showed stacks of pinworm eggs which are D-shaped with double contoured hyalinized shell. Eggs are also surrounded by many large foreign body-type giant cells and granulomas [Figures and ]. Thus, the deposition of eggs by the female gravid worm in the anal wall penetrating through the breaches elicited a severe foreign body type of granulomatous reaction. In the end, the anal canal growth turned out to be a “E. vermicularis worm granuloma with eggs.” This rare interesting case is presented here to stress the fact that worm granuloma can mimic a pseudo tumor in the anal region if untreated for a longer duration.


A 6-month-old female child presented with a progressively increasing lumbosacral swelling which was present since birth []. The child was a full-term normal vaginal delivery and had normal developmental milestones. There was no history of CSF leak or meningitis. The patient had a weakness in ankle movement in both the lower limbs since birth. No obvious history of dribbling or urinary incontinence was forthcoming. On physical examination, there was a soft lumbosacral mass measuring 14 cm × 12 cm × 10 cm in size, obliterating the intergluteal cleft and with normal skin on its surface. The swelling was partially transilluminant and increased in size when the child cried. There were no nevi, hypertrichosis, skin defect, sinus or CSF leak. The head circumference was 44 cm and the anterior fontanelle was lax. The anterior abdominal wall and the genitalia were normal. The anus was patulous.
Computed tomography and magnetic resonance imaging (MRI) revealed a giant terminal lipomyelocystocele [Figures and ] which contained a trumpet like flaring of the central canal of the distal spinal cord and a meningocele located around the dilated central canal, which bulged into the subcutaneous region along with subcutaneous fat tissue. Associated features of partial sacral agenesis in the form of absent dorsal and left sided elements were noticed. Syrinx was seen traversing the whole of the spinal cord. At surgery lipoma infiltrating, the myelocystocele was seen. Lipoma was excised. Caudal portion of trumpet shaped myelocystocele was resected and the remaining base was repaired using 6–0 monocryl suture. Dura was then closed with 4–0 silk suture.
Post-operatively the child had wound infection, which required secondary suturing. The child recovered well with no post-operative deficits. Histopathology confirmed the findings of meningeal tissue with adipose and few wavy nerve fibers [].


A female patient aged 22 years with no cardiovascular history was admitted to the hospital for ATH of 225/150 mmHg. The patient had suffered from intense headaches, palpitations, sweating, excessive nervousness, psychomotor agitation, and mood disturbances, as well as rapid weight loss 10 days before going to the hospital.
Evaluation at admission showed a regular sinus tachycardia at 195 bpm demonstrated by electrocardiogram, a fever of 38.5°, exophthalmia, and a firm, homogeneous diffuse goiter with thrill. A whole-body CT scan showed five tissue and hypervascular masses of 26–30 mm in diameter, two bilateral adrenal, and three retropancreatic para-aortic masses [Figures and ]. Thyroid scintigraphy showed homogeneous increased uptake involving the whole thyroid gland. The highly probable diagnosis of pheochromocytoma was confirmed by the assay of urinary metabolites: normetanephrines = 112,126 nmol/24H (normal range: 300 to 2200), metanephrines = 1602 (normal range: 200 to 1500), and 3 orthomethyldopamine = 176,00 (normal range: 200 to 1400). Biological hyperthyroidism was evident with TSHus < 0.05 μUI/ml (normal range: 0.25 to 5), T3 = 24.37 pmol/l (normal range: 4 to 8.3), and T4 > 70 pmol/l (normal range: 9 to 20). Moreover, the biological workup showed glycemia at 6.60 μmol/l, urea at 7.83 μmol/l, serum creatinine at 74 μmol/l, and serum potassium at 3.8 mmol/l.
Treatment in intensive care consisted of the simultaneous administration of nicardipine via electric pump syringe in decreasing doses of 4 mg/h, then 3, 2, and 1 mg/h of propranolol in intravenous bolus injections of 0.1 mg using an iterative technique, intravenous methylprednisolone dosed at 15 mg/kg and synthetic antithyroid drugs (carbimazole) at the dose of 60 mg/day, but no preoperative treatment preparation by (iodine) have been performed. Surgical excision of the tumors was done by laparotomy after 7 days and normalization of blood pressure and cardiac rate. It was conducted under general anesthesia using propofol, sufentanil, and cistracurium with invasive monitoring of arterial blood pressure and central venous pressure. The surgery was marked by several bouts of ATH and tachycardia, treated with a bolus of nicardipine and propranolol at 1 and 0.1 mg, respectively.
The surgical procedure consisted of the excision of five tumoral masses without adrenalectomy. The postoperative period was uncomplicated, allowing the discharge of the patient on the 5th postoperative day with no treatment beyond the synthetic antithyroid drug. The anatomic pathology examination confirmed the diagnosis of benign multiple pheochromocytoma.


A 12-month-old boy presented with painless progressive swelling of the right scrotum of 1 month duration. There was no history of trauma, fever, weight loss or any constitutional symptoms. The general physical examination revealed an active playful child with normal developmental milestones. On local examination, a right scrotal mass of size 3.5 cm × 4.5 cm, non-tender, firm to hard in consistency with smooth surface and regular margin was felt just above and separately from the testes. However, mobility of the testis appeared to be restricted. Overlying skin was normal and there was no inguinal lymphadenopathy. The abdominal examination was unremarkable.
An ultrasonography (USG) revealed a large heterogeneous mass lesion in the right hemiscrotum, separately seen from the right testis. The chest radiograph was normal and computed tomography (CT) scan of the abdomen and pelvis did not reveal any retroperitoneal lymphadenopathy. There was no abnormal osteoblastic activity on bone scan. The serum alpha fetoprotein (AFP) level was found to be markedly elevated for age (555.1 ng/ml) with normal beta human chorionic gonadotrophin (HCG). Through an inguinal incision, the tumor mass (which appeared to be slightly adherent to the testes) along with the testes was delivered with an intact tunica vaginalis. The spermatic cord was found to be congested. High inguinal orchiectomy was done []. The gross sectioned specimen showed the paratesticular location of the tumor with normal tunica albuginea []. The histopathological examination revealed a tumor arranged predominantly in glandular-alveolar pattern with formation of Schiller-Duval bodies with occasional solid and papillary pattern with hyperchromatic nucleus and moderate amount of clear cytoplasm [] suggestive of yolk sac (endodermal sinus) tumor without any other germ cell component. The testis, spermatic cord and resection margins were free of tumor.
At 18 months follow-up, the patient was doing well with AFP level of 5 ng/ml and no radiological evidence of metastasis.


A 45-year-old female patient presented to our department with a chief complaint of pain in the left molar region of the mandible for 1 month. The patient was otherwise healthy, and her physical examination showed no significant abnormality. Intraoral examination revealed a carious left mandibular first permanent molar and a missing second permanent molar which was extracted due to caries 2 years ago.
Intraoral periapical radiograph showed well-defined irregularly shaped sclerotic masses corresponding to the roots of first and second permanent molar teeth of left mandible []. Orthopantomogram showed multiple well-defined sclerotic masses with radiolucent border in both right and left molar region of the mandible []. These sclerotic masses were surrounded by a thin radiolucent border and appeared to be unattached to the root apices. All teeth were vital. The results of routine hemogram and serum alkaline phosphatase were within normal limits. Biopsy was not done as the case was diagnosed as FCOD on the basis of characteristic clinico-radiological features.


A 36-year-old female patient had complained of a swelling in the right side of the tongue since 1 month, which had started spontaneously and then showed a rapid increase in size. The patient developed a dull and intermittent pain 2 days after the swelling had started. She had difficulty with speech and swallowing. Extra-orally, a single submandibular lymph node on the right side was palpable, tender, soft in consistency and freely movable. Intra-oral examination revealed a diffuse swelling on the right side involving the dorsum, right lateral border [] and ventral surface of the tongue []. It extended from the tip of the tongue to 1 cm beyond the anterior two-thirds. Medially, it crossed the midline by 2 cm. The overlying mucosa was normal and the surface of the swelling was smooth; margins were ill-defined. There were no ulcerations or sinus openings. On palpation, the swelling was tender and firm in consistency and moved along with the tongue movements.
A provisional diagnosis of malignant soft tissue neoplasm from the muscles of the tongue or minor salivary glands was made.
Magnetic resonance imaging (MRI) neck plain revealed a well-defined T1 hypo-STIR [] hyperintense lesion involving the anterior half of the tongue with displacement of the intrinsic muscles superiorly and to the left side. The lesion showed small GRE/T2 [] hypointense foci within the lesion.
Excision biopsy was done under general anesthesia and the histopathological findings revealed isomorphic basaloid tumor cells with a cribriform pattern and numerous cystic spaces []. There was compressed dense connective tissue in the periphery that was infiltrated by tumor islands. Few areas showed perineural invasion. Correlating the clinical, MRI and histopathological findings, a final diagnosis of adenoid cystic carcinoma of mobile tongue was made.


A 38-year-old G1P0 primigravida was admitted to our hospital at 40+6 weeks gestation for a normal vaginal delivery. She had no history of medical illness or trauma. The initial fetal heart rate (HR) was 120-150 beats/min and showed good reactivity and variability. When the diameter of the cervical opening was approximately 4 cm, lumbar epidural analgesia was planned, and an intravenous (IV) infusion of lactated Ringer's solution (1,000 ml) was administered. An 18-gauge Tuohy needle (Portex® Regional Anaesthesia Tray, Smiths Medical, UK) was blindly inserted into the lumbar epidural space at the L3-4 level in the left decubitus position. A Tuohy needle was advanced to 5 cm, at which point the epidural space was identified on the first attempt using the loss of resistance (LOR) technique. After ensuring that there was no cerebrospinal fluid (CSF) or blood leak, we slowly injected a test dose, i.e., 4 ml of 1% lidocaine with 15 µg of epinephrine. An epidural catheter (Portex® Regional Anaesthesia Tray, Smiths Medical, UK) was inserted without any resistance. The catheter tip was threaded 4 cm in the cephalad direction beyond the Tuohy needle, after catheter placement. We confirmed that there was no CSF and blood by aspirating the catheter. Then, we mixed 20 ml of 0.75% ropivacaine and 100 µg of fentanyl with normal saline (total volume, 100 ml), and subsequently injected 12 ml of the mixture, The rest of the mixture was injected into the epidural space with a patient controlled epidural analgesia (PCEA) pump (Pain Management Provider®, Abbott, USA) with a basal flow rate of 5 ml/h plus a 5 ml bolus with a 5 min lockout. At 5 minutes after the initial dose administration, her blood pressure (BP), HR, and respiratory rate (RR) were 120/70 mmHg, 72 beats/min, and 20 breaths/min, respectively. We subsequently assessed the extent of bilateral sensory blockade by observing the loss of cold sensation to alcohol sponges and the loss of sensation to pinpricks. At that time, the sensory level was located at the tenth thoracic dermatome, and the motor function was intact. At 15 minutes after the initial dose administration, the patient complained of a pulling sensation and tightness at the back of her neck. At 20 minutes after the initial dose administration, the patient showed drowsiness and a depressed mental status; therefore, we encouraged her to take deep breaths and stopped the PCEA pump. The total injected volume of the drug mixture was 1.5 ml. At that time, her vital signs were as follows: BP, 90/60 mmHg; HR, 60 beats/min; and RR, 8 breaths/min. The sensory level was at the eighth thoracic dermatome; however, no change in motor function was observed. Because of the reduced RR and depressed mental status, the patient was placed in the Trendelenburg position and was administered 100% O2 via a facial mask at a flow rate of 5 L/min. At 3 min after position change, her vital signs were as follows: BP, 76/49 mmHg; HR, 55 beats/min; RR, 8 breaths/min; and SpO2, 95%. The sensory level was the third thoracic dermatome, but her motor functions were not affected. After the patient was injected with 10 mg of ephedrine, an obstetrician examined the fetal status via ultrasonography. The fetus showed severe bradycardia, (HR, 70 beats/min); despite being in the normal range (HR, 100-150 beats/min) before this event. Therefore, we decided to perform an emergency cesarean section, which was planned to occur within 30 min, followed by prompt intrauterine fetal resuscitation. At that time, the patient's vital signs were as follows: BP, 80/51 mmHg; HR, 52 beats/min; RR, 8 breaths/min; and SpO2, 92%. We moved the patient to the left lateral recumbent position, injected 0.5 mg of atropine via the maternal IV line, started rapid fluid replacement, and continuously monitored the fetal HR. At 5 min after intrauterine fetal resuscitation, the fetal HR was 100 beats/min, but the patient still showed a depressed mental status and complained of dyspnea. Artificial respiration was performed immediately by using AMBU bag because the patient's SpO2 decreased to 82%. Consequently, her SpO2 recovered to 95%, while the fetal HR increased to 120 beats/min and showed good variability. The fetal HR had decreased for approximately 10 minutes. Fifteen minutes after recovery of the SpO2 level, the patient was transferred to the operating room on artificial ventilation. The arterial blood gas analysis performed immediately before the operation revealed the following: pH, 7.086; PaCO2, 66.6 mmHg; PaO2, 218.6 mmHg; HCO3, 19.6 mEq/L; base excess, -10.8; and SaO2, 99.0%. The patient's preoperative vital signs were as follows: BP, 94/62 mmHg; HR, 90 beats/min; and RR, 8 breaths/min. Anesthesia was induced by the administration of 4 mg/kg of IV thiopental, while tracheal intubation was facilitated by the administration of 1 mg/kg of IV succinylcholine. After tracheal intubation, anesthesia was maintained with O2, air, and 1.5-2.0 vol% sevoflurane. For the muscular block, 4 mg of vecuronium was administered. A live infant, weighing 3.06 kg, was delivered. The incision to delivery time was 3 min. The Apgar score was 8/9. After 40 minutes, the patient was transferred to a recovery room after extubation, because she showed complete recovery during the emergence period. After that point, her vital signs were stable, tidal volume > 400 ml, RR was 12 breaths/min, and SpO2 was 99% in room air. A neurological examination performed in the recovery room, showed that the patient had an alert mental status, and that her orientation-related mental functions were normal. The sensory and motor functions of the patient recovered completely, but she complained of mild numbness in her thighs and calves. Arterial blood gas analysis performed in the recovery room revealed the following: pH, 7.350; PaCO2, 30.7 mmHg; PaO2, 156.3 mmHg; HCO3, 16.6 mEq/L; base excess, -7.8; and O2 saturation, 98.9%. After 1 hour in the recovery room, the patient was transferred to the general ward. After 2 days, no post-dural puncture headache or other neurological complications were observed, therefore, she was discharged along with the infant, who did not show complications, or neurological abnormalities.


A 71-year-old female visited the outpatient department with a 2-week history of coughing with sputum and dyspnea on exertion. She had no other medical history with the exception of pulmonary tuberculosis at the age of 30 years. After administration of anti-tuberculosis medication, she was completely cured.
A chest X-ray performed during the patient's first visit to the outpatient department showed consolidation in the right lower lung field and focal consolidation in the left lower lobe. Sputum acid-fast bacteria (AFB) staining was negative. On bronchoscopy, the inlet of the right lower lobe was crushed out, especially the superior segment of the lower lobe. However, intraluminal lesions were not found. Based on these results, the patient was diagnosed with community-acquired pneumonia and was prescribed clarithromycin for 7 days. She was scheduled for a follow-up chest X-ray after finishing the medication, but she did not return to the hospital.
The patient returned to the hospital after 6 months because of dyspnea on exertion (American Thoracic Society grade 2). A chest X-ray was again performed (). The right middle lobe and lower lobe showed increased haziness and density together with linear consolidation in the left lung. A chest CT scan was then performed (). Consolidation was seen in all right lung zones with the exception of the superior segment of the right upper lobe. The right middle and lower lobes were mainly collapsed. Both the upper lobe and left lower lobe had many nodules. They also showed GGOs. Compared with the first chest X-ray, increases in the extent and haziness of the lesions were observed. Therefore, due to suspicion of pneumonia aggravation, we administered cefepime and clindamycin after performing a culture. However, we could not rule out the possibility of pulmonary tuberculosis reactivation or a cancerous mimic of pneumonia. Therefore, sputum AFB staining and cytology were performed, followed by bronchoscopy. Transbronchial lung biopsy was attempted, but failed. The AFB stain and cytology tests were all negative, and there was no endobronchial mucosal lesion. Upon admission, vital signs were as follows: blood pressure 100/60 mm Hg, heart rate 114 beats per minute, respiratory rate 20/min, and body temperature 39.1℃. The patient was alert and oriented, but showed an acutely ill-looking appearance, and there were rales in the right lower lobe zone with a rough respiratory sound.
Peripheral blood test results were as follows: white blood cells 25,200/µL (neutrophils 86%, lymphocytes 7.3%, monocytes 6.1%, eosinophils 0.5%, and basophils 0.3%), hemoglobin 14.6 g/dL, and platelets 205,000/µL. In addition, the C-reactive protein level increased to 6.6 mg/dL. Chemistry test results were as follows: aspartate aminotransferase 20 IU/L, alanine aminotransferase 15 IU/L, total bilirubin 1.4 mg/dL, lactate dehydrogenase 440 IU/L, total protein 7.1 g/dL, albumin 3.8 g/dL, blood urea nitrogen 12 mg/dL, and creatinine 0.6 mg/dL. The results of arterial blood gas analysis at room temperature were as follows: pH 7.46, pCO2 31 mm Hg, pO2 67 mm Hg, HCO3 22 mmol/L, and saturation 94%. There was no improvement on chest X-ray after finishing the antibiotics or after changing them. The bronchoscopic washing cytology test results obtained before admission were suspicious for malignancy. Thus, a peripheral blood carcinoembryonic antigen level was obtained, and the result was 0.8 ng/mL.
For a definitive diagnosis, an open lung biopsy was performed in the basal segment of the right lower lobe. The lesion in the right lobe showed a homogenous nodular pattern during the surgery, and the biopsy was extracted from the right basal segment. A 2.5×1.7-cm section of tissue was extracted for frozen biopsy and was diagnosed as AAH (). Regular biopsy using the rest of the tissue after frozen biopsy revealed AAH with a negative epidermal growth factor receptor (EGFR) mutation test. The blood and sputum cultures upon admission were both negative, as was the AFB culture of sputum. The patient's fever and other symptoms showed improvement after 26 days of hospitalization with supportive care. The patient was then finally discharged.
After 4 months, the patient was admitted again due to aggravated dyspnea on exertion. Both a chest X-ray () and CT scan () were performed. On the chest CT scans, the right lung consolidation was improved, but the extent of the GGO had increased. In the lateral segment and posterior basal segment of the left lower lobe, the GGO pattern as well as the extent and haziness of the consolidation had increased. Moreover, the size of the multiple nodules with GGO increased. Thus, the patient was diagnosed with AAH in progression. According to these findings, positron emission tomography-CT (PET-CT) was performed to rule out malignancy and in preparation for possible chemotherapy. PET-CT was performed on the first day. The images in show that there was some glucose uptake in the lesions in the collapsed part of the right lower lobe, but no uptake in the regional lymph nodes. These observations were believed to be secondary to an infection rather than a cancerous lesion. The biopsy results were consistent with AAH. Gefitinib was administered to the patient after 7 days of hospitalization. After 17 days of treatment with gefitinib, a follow-up chest X-ray was performed. shows that the right lung and hazy left lower lobe consolidation had improved compared with the findings before gefitinib treatment.