An 18-year-old woman, with a 3-cm superficial lump of the right breast, underwent a surgical excision in January 2011 at another Institution, with a diagnosis of high-grade angiosarcoma. She had no remarkable family or medical history. Menarche had occurred at age of 14 years, with normal regular periods. In February 2011, she was referred to the Humanitas Research Hospital, Milan, for further work-up. After staging procedures, a right radical mastectomy was carried out with no evidence of residual disease at the pathological examination. No adjuvant treatment was delivered.
In October 2011, a positron emission tomography/computed tomography (PET/CT) scan revealed a pathologic uptake in the right axilla and the patient underwent an axillary dissection. The pathological examination was consistent with lymph node metastasis of high-grade angiosarcoma. From November to December 2011, adjuvant chemotherapy with doxorubicin 25 mg/m2/day and ifosfamide 1 g/m2/day (both on days 1–3) every 21 days was administered. During treatment, the patient reported menstrual irregularities but no amenorrhea.
In January 2013, a nodule of the right thoracic wall was surgically removed confirming a relapse of angiosarcoma. Adjuvant radiation therapy was proposed, but the patient refused it.
In September 2013, a PET/CT scan revealed a further local relapse along with the involvement of the contralateral axillary lymph nodes for which our patient underwent chemotherapy with gemcitabine 1000 mg/m2 on days 1, 8 every 21 days for 6 cycles, achieving a partial response according to RECIST criteria, followed by surgery.
In May 2016, for progressive disease with distant pleuropulmonary metastases and further local recurrence, the patient received pegylated liposomal doxorubicin 50 mg/m2 on day 1 every 28 days, with stable disease after 3 cycles and disease progression after 6 cycles. Therefore, in November 2016, pazopanib at a dose of 800 mg daily was started. Thyroid function tests at the baseline were within the normal range (thyroid-stimulating hormone [TSH] 2.25 mIU/l with normal range of 0.27–4.20). In December 2016, thyroid monitoring showed an increase in TSH (22.83 mIU/l), a decrease in fT3 1.8 pg/ml (normal range: 2.0–4.4) and normal fT4 10.6 pg/ml (normal range: 9.3–17.1). Anti-thyroglobulin and anti-thyroid peroxidase antibodies were absent. A thyroid ultrasound excluded the presence of nodules or other abnormalities. Levothyroxine replacement therapy was started at a dose of 50 μg daily. In January 2017, secondary amenorrhea occurred. Beta-HCG dosage was negative (2.5 mIU/ml, n.r. 0–5.3). In February 2017, thyroid tests showed a stable TSH level (22.87 mIU/l), and a normalization of fT3 (2.5 pg/ml). Dose of levothyroxine was increased to 75 μg daily. A PET/CT scan showed a complete response of the lung lesions and a partial response of the local relapse. In March 2017, while still on therapy with pazopanib, normal thyroid function was restored (TSH 4.21 mIU/l, fT3 3 pg/ml, fT4 11.9 pg/ml) and reproductive hormone levels were as follows: follicle-stimulating hormone (FSH) was 48 mIU/ml, luteinizing hormone (LH) was 75.8 mIU/ml, and estradiol (E2) was 61 pg/ml. Anti-Müllerian hormone (AMH) was 3.27 ng/ml (within the lower values of the normal range: normal range: 0.96–13.34 for 18–25-year-old women, 0.03–3.27 for 41–45-year-old women). Antral follicle count (AFC) obtained by trans-vaginal ultrasound revealed a right AFC of 4 and a left AFC of 2 (normal count: 7).[ Based on these data, we diagnosed a primary ovarian insufficiency. In September 2017, a PET/CT scan showed a local progression of disease, pazopanib was stopped and a new treatment with paclitaxel was proposed. After discussion with the patient about possible treatment-related adverse events, patient's refusal of hair-loss inducing therapies and clinical benefit from gemcitabine previously administered, a further line of treatment with gemcitabine was started in October 2017. In November 2017, after 2 months from pazopanib discontinuation, menses returned regularly. Hormonal assays showed FSH 9.2 mIU/ml, LH 10.6 mIU/ml, E2 120 pg/ml, in the follicular phase.
In April 2018, the patient experienced further disease progression and paclitaxel-based treatment has been proposed. The patient has provided a written informed consent for the case details to be published; approval from the ethical committee was not necessary, in line with the regulations of our Institution.


A 68-year-old man presented to the emergency department (ED) with severe right thigh pain and left leg spasms after having suffered a ground-level fall onto his right side. On physical exam, he was hemodynamically stable, with minor skin abrasions on his right elbow and both lower extremities. The pelvic X-ray showed a displaced comminuted right intertrochanteric fracture (Figure ). His right lower extremity was placed in a Hare traction splint, and he underwent an open reduction and intramedullary gamma nail fixation of the right femur. He was given baclofen (10 mg twice daily) and diazepam (10 mg twice daily) for his left leg spasms postoperatively.
With regards to his past medical history, he reported a 20-year history of stiff-person syndrome (SPS) with frequent muscle spasms and recurrent falls. He stated his doctors have been unable to adequately control his symptoms of SPS despite multiple therapies. On initial diagnosis, he was started on baclofen and diazepam with no significant relief of his spasms. He then underwent IVIg treatments three days per month for one year, followed by diazepam as maintenance therapy. He reported a mild improvement in symptoms with IVIg infusions but they did not sufficiently improve his ambulation or minimize his risk of fall. He continued to have falls, especially during physical activities such as playing tennis; one of which caused a subdural hematoma that required a left craniotomy. He remarked that he was once a high-functioning, active individual but has since been limited by his condition.
On postoperative day one, he reported that his right thigh pain was declining, but he continued to have painful left leg spasms that worsened with movement. On physical exam, he displayed significant gait instability, decreased functional mobility, hypertonicity of his bilateral upper extremities, and prominent muscle spasms of his left quadriceps femoris muscle. His neurological examination was otherwise within normal limits. The patient’s pain from the fracture continued to improve over the second and third postoperative days.
Despite adequate recovery from the surgery, the patient continued to have marked hypertonicity of his upper extremities and painful muscle spasms of his left leg that prevented him from participating in PT. Anti-GAD antibody levels were remarkably elevated at a value >250 IU/mL (0.0 - 5.0 IU/mL) that was too high to be quantified by the laboratory. Creatine kinase (CK) levels were elevated at 469 U/L (35 - 232 U/L).
Due to his nonresponsiveness to conventional therapy and the high severity of disease, a series of seven treatments of TPE over 14 days were initiated. While discussing the treatment plan with the patient, he expressed little hope in TPE improving his condition, as he had found minimal relief after multiple trials of medications, including the year-long trial of IVIg therapy.
Over the two-week course of TPE, his functional status and symptoms of SPS significantly improved. Although his hyperreflexia remained stable at 3+, his muscle spasms and hypertonicity progressively improved after each treatment of TPE to the point that he had normal range of motion in all extremities. While receiving TPE, he was able to tolerate three hours of PT sessions daily for up to five days per week, whereas he had zero rehabilitation potential before TPE was initiated. He made significant functional gains using adaptive equipment, corrected his gait, and was eventually able to ambulate without assistance. He felt his symptoms were adequately controlled for the first time since his diagnosis and reported having no symptoms by hospital day five. Shortly after his TPE therapy was complete, he was deemed to be in a stable condition and discharged to inpatient rehabilitation.


This case report will focus on a 32-year-old homeless male with a long history of alcohol abuse with multiple relapse episodes. He states that he is currently abstinent and had recently been started on Acamprosate by his primary care physician. He is living in a shelter with his girlfriend who had just recovered from an infection of her bilateral axilla. The patient stated that he used his girlfriend's deodorant which led to the development of large, painful boils under his right axilla and groin simultaneously. After taking a warm bath he self-lanced both boils and later that day he began to experience fatigue, muscle aches, pains, and fevers. Later that night he took Acamprosate with an energy drink. Shortly after taking it he developed severe urticaria and pruritis. He went to bed and the next morning he developed significant angioedema of the bottom lip as shown in Figure , below.
The patient went to an urgent care center, was treated with Prednisone, and had a nasal swab for influenza which was positive. He was treated with Tamiflu and sent home. He went on to develop significant angioedema of the upper lip as seen in Figure below.
Due to the persistent and severe nature of the symptoms the patient decided to go to the emergency room. Acamprosate was immediately discontinued and the patient was informed to never take this medication again. He was treated with steroids and Benadryl with gradual improvement of symptoms until complete resolution.


This study was approved by Ethics Committee of Dalian Maternal and Child Health Hospital, and the informed consent signed by the patient.
The patient was female, was 37 years old, was from Liaoning province in China, and had been examined at our hospital at 13 weeks of pregnancy. The color ultrasound results showed that the nuchal translucency (NT) value was 2.4 mm, and NIPT using an Ion Proton sequencer (CapitalBio Technology Inc., Beijing) at 16 gestational weeks was performed. However, the Z-score(102.835) for Chr8 was outside the normal range and suggestive of trisomy 8. Because most cases of complete trisomy 8 are spontaneously aborted in the early stages of pregnancy, we highly suspected that trisomy 8 was from the mother and that trisomy 8 was mosaic. Further examination by three-dimensional color ultrasound suggested a single live fetus with no abnormalities. The patient denied smoking, drinking or radiation, and was without chemical exposure. We evaluated the brain development and reproductive system of the fetus by ultrasound, there was no obvious abnormal clinical manifestations, and the fetus had developed normally. The patient had previously been pregnant three times, resulting in one cesarean section, one spontaneous abortion, and one birth to a girl who is currently 8 years old and in good health.
The peripheral blood from the patient was extracted for hemocyte classification analysis and smear microscopy detection. We found no abnormal juvenile cells in the peripheral blood by smear microscopy. Through the detection of leukocyte immune typing, we found that CD117+/CD34+ cells accounted for 0.30% of nuclear cells, that these cells were all normal myeloid primitive cells, and that granulocytes accounted for 41.46% of nuclear cells. Immature monocytes occupied 0.02% of nuclear cells (0.53% of monocytes). Bone marrow stem cells, B cells, T cells, and NK cells were normal, and no abnormal myeloid primitive cells (< 10− 4) were observed in this clinical report. In addition, the development model for cells was normal (Tables and ).
Then, the peripheral blood from the patient, her husband and her daughter and amniotic fluid from the patient were collected to undergo chromosome G band analysis. From these results, we knew that the peripheral blood karyotype of the patient was 47,XX,+ 8[67]/46,XX, while the amniotic fluid karyotype was 46,XX; the karyotypes for her husband and daughter were 46,XY, and 46,XX, respectively. In addition, trisomy 8 mosaicism was confirmed using Ion Proton sequencer (CapitalBio Technology Inc., Beijing) at 400 flows according to the manufacturer’s instructions. The amniotic fluid, pharynx, cheek, saliva and cervical abscission cells samples from the patient were undergone next generation sequenced. The retained reads were aligned to the human genomic reference sequences (hg19) using the BWA. The fetal DNA concentration was calculated as a quality control, as described in Yin’s paper []. Combined GC-correction and Z-score testing methods were used to identify fetus autosomal aneuploidy for trisomy, as described in Liao’s paper []. A Z score range from − 3 to 3 was considered to indicate a low risk for a trisomy chromosome, and if Z score was > 3, the sample was in the high-risk zone. The result was considered as a trisomy when the Z-score was far higher than the normal value and when the average score of the black curve was in + 20. When the Z-score was far below the normal value and when the average score of the black value curve was − 20, a monomer was considered. Chromosomal chimerism was considered 1) when the Z-score was greater than or less than plus or minus 3 but far less than the Z-score of complete trisomer or greater than the Z-score of the complete and 2) when the mean score of the black curve was greater than or less than 0 but far less than the mean value of the complete trisomer or greater than the mean value of the complete monomer. Chimeric proportion (%) = Z-score range /0.2/100 (female) or Chimeric proportion (%) = Z-score range /0.2/100/concentration of the sample (male). Moreover, NGS result suggested that the proportions of trisomy 8 in different tissues were obviously different (Fig. ). The proportions for trisomy 8 in cheek, saliva and cervical abscission cells samples were 5, 60, and 100%, respectively. In addition, the amniotic fluid was 0%, which suggested that the fetus was normal (Fig. ). Last, the chromosomes of the patient and her baby were further examined using chromosome microarray analysis (CMA) with the higher resolution CytoScan® HD platform (Affymetrix, Santa Clara, CA) (Fig. ). The CMA analysis showed that the chr.8 detection result in the plasma of the patient was arr[GRCh37](8) × 3,11p15.5p13(230750–33,455,733) × 2 hmz, and the result of her baby was normal.


A 62 year-old-woman, complaining from micturition lipothymia for six months, was admitted in our hospital. She had no significant past medical history, and there was no history of headaches, palpitations, profuse sweating, hypertension or gross hematuria. Her family history was unremarkable.
Physical examination was normal, and blood pressure was within normal range at 120/80 mmHg. Cytobacteriological examination of the urine was negative. The patient was not anemic and the white cell count was within normal range. There was no renal failure.
An abdominal ultrasonographic examination was performed initially, which depicted an approximately 40 mm heterogeneous mass along the anterior wall of the bladder with abundant internal flow at Doppler interrogation ().
An abdominal computed tomography (CT) scan, with intravenous administration of contrast medium, revealed a 32 mm heterogenous lobulated anterior bladder wall mass. The mass showed intense uniform enhancement with contrast study. There was no sign of any lymphadenopathy or metastatic invasion ().
Further imaging with Iodine-123-meta-iodobenzylguanidine (I-123 MIBG) scintiscan confirmed the presence of bladder pheochromocytoma without metastatic disease ().
The 24-hour urine metanephrine and normetanephrine measurement was within normal levels.
The patient underwent a complete and deep transurethral resection of the bladder tumor. A solid submucosal mass was seen on the left lateral wall near the neck of the urinary bladder, with normal mucosal covering. The resection was complete and tissue was submitted to histopathological examination. During the procedure, the patient became severely hypertensive. Her blood pressure raised up to 236/118 mmHg and pulse rate dropped to 46/min. This episode was controlled with intraoperative intravenous antihypertensive and atropine. Postoperative recovery was uneventful.
The histological examination of the tumor confirmed the diagnosis of vesical paraganglioma showing tumorous small cells with a positive immunostaining for synaptophysin and anti-CD 56 ().
The patient underwent partial cystectomy (). The surgical margin of the removed specimen was negative. Postoperative recovery was uneventful.
On a recent follow-up at 12 months, her cystoscopy examination did not reveal any evidence of recurrence. Her radiological assessment (thoracic-abdomino-pelvic CT) was negative for metastatic disease.


At our institute, a 58-year-old female patient in good health came to our attention. Access to our department was determined by a persistent abdominal pain localized in the right lumbar region. The collection of anamnestic data revealed a small cell type NEC, discovered two years before, with positivity to Synaptophysin and Neuron Specific Enolase (NSE) but not to Chromogranin A, of the left ethmoid-nasal-orbital region, treated with chemo- and radiotherapy cycles. In our department the patient underwent a total body Computed Tomography (CT) scan () which showed the presence of a voluminous mass in the right retroperitoneal area, with a maximum transverse diameter of 12 cm, presenting areas of necrotic colliquation in its context. This mass infiltrated the sixth and seventh hepatic segments, dislocated and infiltrated the inferior intrahepatic vena cava, dislocated the right kidney by infiltrating the superior pole and extended to the right lumbar region. This mass also infiltrated the anterior abdominal wall in the right lumbar region, determining an edema of the underlying subcutaneous adipose tissue, and dislocated, but did not infiltrate, the cephalic portion of the pancreas. Surgery was performed with total median laparotomic access. The mass was removed en bloc (, 3).
The whole mass weighed 1200 g, and was 17 × 13 × 12 cm in size, linked to all removed structures (, 5).
The final histological examination showed a NEC composed of small cells with immunohistochemical characters that were Synaptophysin-positive, NSE positive, but chromogranine negative ().
The post-operative course was satisfactory and the patient was released from the hospital on the 24th day in good condition and entrusted to oncologists for treatment of the case. The patient had a positive follow-up at 6 months and 12 months.


A 62-year-old Caucasian male with a medical history significant for squamous cell laryngeal cancer in remission, history of tracheostomy, type 2 diabetes mellitus, and hypothyroidism presented to an outside institution with a 3-day history of left ear pain with associated nausea and vomiting. Initial laboratory studies demonstrated elevated nonspecific acute phase reactants. Computed tomography (CT) indicated left middle ear swelling and enhancement of the left soft palate. Fungal cultures were negative. A diagnosis of left malignant otitis externa was made, and appropriate intravenous and topical antimicrobial therapies were initiated. Due to clinical deterioration, the patient was transferred to our institution where initial neurological assessment showed a full left side facial droop, left otic vesicles on the pinna, and gait ataxia. Repeat CT scan was consistent with malignant otitis externa. On hospital day two, the patient's condition continued to worsen and neurological exam indicated left cranial nerve (CN) VI and VII nerve palsies, horizontal diplopia of the left eye, conductive hearing loss, skew deviation, and persistent herpetic lesions on the hard palate and the outer ear (). There was no nystagmus, and clinical head impulse exam was negative.
Due to suspected multiple CN involvement, contrast brain magnetic resonance imaging (MRI) was conducted followed by a lumbar puncture (LP). Brain MRI showed a hyperintense lesion along the left middle and inferior cerebellar peduncle along the fourth ventricle (Figures –). Cerebrospinal fluid (CSF) revealed elevation in glucose, protein, and nucleated cells (lymphocyte-predominant). VZV quantitative polymerase chain reaction (PCR) tested positive. Herpes simplex virus (HSV) 1 and 2 titers tested negative (). Diagnosis of Ramsay Hunt syndrome (RHS) with concomitant VZV encephalitis was made at this time.
Initial treatment included empiric antibiotic and antifungal medications; however, after the confirmation of viral etiology, these were discontinued. Intravenous acyclovir 800 mg 5x/day and prednisone 70 mg oral daily was initiated. One day later, the patient showed significant improvement in visual deficits. Mild lateral gaze palsy remained along with full left facial weakness, gait ataxia, and vesicular lesions on the hard palate and ear. He remained in the hospital for 9 days and was discharged on acyclovir 20 mL 5x/day for 14 days.
At 4-month follow-up, the patient demonstrated improved balance but had persistent left facial droop and left-sided hearing loss. The remainder of the neurological exam was unremarkable. Due to the recurrence of left otic discharge, the patient was discharged on a 10-day course of 1 g valacyclovir three times/day as an outpatient.


A twenty-two-year-old Iranian woman arrived at our institute with a complaint of severing back pain at the level of T5-T6 vertebrae since two months ago. There was no definite initiating cause and the pain was not associated with the activity. The pain did not radiate into any other part. She did not consume alcohol or illicit drug and had no history of underlying diseases.
Laboratory data were all within normal ranges except slightly elevated ESR.
The radiological study was performed. The Thorax spiral CT scanning without contrast injection was done and demonstrated an expansible bony lesion involving posterior arc of the right 6th rib, costovertebral joint and a portion of the T6 vertebrae. It was measured about 68*44 mm. In MRI, Right D5, D6, and D7 paravertebral mass were seen. The intervertebral foramen was also involved by the enhanced lesion ().
Then CT-guided biopsy and fine needle aspiration of the lesion was taken. A lobulated and irregular margin without cortical destruction was present. Macroscopic appearance of lesion was suggestive of benign osseous mass but extension into the body and right side of posterior elements of the adjacent vertebra was also seen. The specimen was sent for further evaluations. It contained multiple tiny pieces of brown soft tissue. The histopathologic finding was suggestive of norm cellular bone marrow. There were no malignant or atypical cell ().
Based on previous cases we decided to perform thoracostomy under general anesthesia by an expert spine surgeon. At the operating room, we performed resection of the proximal parts of right 6th rib and partial T5 and T6 corpectomy. During the surgery, we completely excised the lesion and a safe margin to avoid local recurrence. Intra-operatively frozen section was sent for rapid analysis to determine the safe margin from the lesion in surrounding parenchyma and minimize tissue damage. Although the mass had only involved the rib, and not the surrounding area, we gently removed reactive parts of the cortex for safety. Thus, intraoperative observation also helped in determining the margin of excision to decrease excessive tissue damage.
Then she was admitted at the Neurosurgery Intensive Care Unit for three days under our close observation. The post-operative period was uneventful. Wound healing occurred without infection or inflammatory response.
During one-year follow-up, she did not experience any abnormality. General physical exams were normal. Laboratory evaluations were all within standard ranges. However, we performed spiral chest and abdominopelvic CT scan; no lesion or any sign of remission was found.
Based on our review on literature, there were seven similar cases of Focal Hematopoietic hyperplasia reported in articles. Here we also compared our findings with their reports and discussed similar features.


Case 1 was admitted with coughing during feeding and respiratory tract infections lasting for 2 months when she was 3 years old. Because of the poor general condition of the case, we decided to manage the patient conservatively. During this period, oral feeding was ceased, and tube feeding and broad-spectrum antibiotics were recommended. We observed spontaneous healing of the fistula after 2 months. She has been on full oral feeding for the last 1 year without any respiratory symptoms.


A 60-year-old man presented with the new onset of an asymptomatic blue tongue. His friends had noted the change in the color of his tongue. He had been eating Cookie Monster simple gelato (Frozen Bean Corporation, Rancho Cucamonga, CA). The dry mix powder consists of not only FD&C blue no. 1 dye, but also several other ingredients: sugar, non-dairy creamer [non-hydrogenated coconut oil, corn syrup solids, sodium caseinate (a milk derivative), sugar, dipotassium phosphate, silicon dioxide, propylene glycol esters of fatty acids, monoglycerides and diglycerides, salt, soy lecithin, carrageenan, artificial flavor and annatto], chocolate cookies based cake {enriched flour [wheat flour, niacin, reduced iron, thiamine mononitrate (vitamin B1), riboflavin (vitamin B2), and folic acid], sugar, palm oil, and cocoa [processed with alkali]}, crystalline fructose, nonfat milk, sweet whey, cocoa powder (processed with alkali), guar gum salt, natural and artificial flavors, silicon dioxide (anti-caking agent).
An examination of his tongue showed that the dorsal surface had a confluent blue color (Figure ). The distal tip and the lateral surfaces of the tongue retained their normal color. In addition, there was subtle blue discoloration of the mucosal upper lip.
The blue appearance of his tongue and upper lip spontaneously resolved within six hours. The dyschromia did not recur. Subsequent investigation determined that the composition of the gelato included FD&C blue dye no. 1.


Case 1 was a 48-year-old female patient with no systemic diseases, the bilateral posterior tooth were loss (Figure ), which seriously compromised the daily mastications. Clinical and radiology examination indicated that the bone quantity of maxillary and left mandibular regions is sufficient for implant placement; however, the bone width at right mandibular region is significantly limited, moreover, the residual alveolar crest shaped as a narrow blade. CBCT scanning revealed that 45, 46, and 47 were missing, the vertical height of alveolar bone was sufficient but the horizontal width of alveolar crest was merely 3-4 mm (Figures and ).
The modified bone-splitting technique with simultaneous GBR was planned at the posterior region of right mandible, and delayed implant placement was designed at 6 months after GBR. Conventional implant prosthodontics were designed for other missing teeth.
After the disinfection and local anesthesia, the linear incision was proceeded along the “blade shaped” top of alveolar ridge by the piezosurgical blade (PIEZOSURGERY® 3; Mectron S.p.A.) on the mesial-distal direction. At the mesial and distal ends of top incision, 2 incisions were proceeded perpendicularly, and another A horizontal incision was cut 10 mm parallel to the top incision, all these 4 incisions penetrated through the cortical bone to reach the cancellous bone and bone marrow (Figure ). Bleeding holes were prepared on the surface of the bone-splitting region. A 4-mm wide osteotome was inserted from the crestal incision and extended into the cancellous bone, the osteotome was gently pushed by bone chisel for approximately 8 mm, and linear bone expansion was processed at buccal-lingual direction (Figure ). Bio-oss bone substitutions were added in the expanded area (Figures and ) and covered by a resorbable membrane (Haiao Heal-ALL, ZH-BIO) (Figure ), a piece of PRF membrane (Figure ) was attached on the top of the resorbable membrane to provide cytokines for bone/soft tissue regeneration. Finally, the surgical region was closed by simple interrupted suture(H-563; Holycon) (Figure ). Patients were instructed to use clindamycin and ornidazole for 7 days to prevent infections; the sutures were finally removed after 14 days.
CBCT imaging at 6 months showed that an average augmentation of 2-3 mm in alveolar ridge width was achieved at surgical site (Figure ). With sufficient bond quantity, conventional dental implantation was performed under local anesthesia, and 3 implants were inserted (#45:3.6 mm(ф) × 10 mm(L), #46:4.5 mm(ф) × 8 mm(L), #47:4.5 mm(ф) × 8 mm(L); Dentium) (Figure ). The final prosthodontics were finished at 3 months after implant placement (Figures and ).
Significant bone width increment was achieved by the combination of modified piezosurgery technique and simultaneous GBR. The bone width was shown to be stable during and after 2 years of final restoration, and no significant bone resorption was revealed (Figures and ).


Case 2 was a 58-year-old woman without systemic disease. The 35, 36, and 37 were missing for the severe dental caries (Figure ). CBCT showed the edentulous region of 35, 36, and 37 had a “blade shaped” residual alveolar ridge which is significantly insufficient for conventional implant placements, the width of residual alveolar crest was merely 3-4 mm (Figures and ).
The modified bone-splitting technique with simultaneous GBR was planned at the posterior region of left mandible, and delayed implant placement was designed at 6 months after GBR.
After disinfection and local anesthesia, the same alveolar ridge augmentation was performed at 35-37 sites as in case 1 (Figures , , , ). After the bone splitting, Bio-oss bone substitution was added into the bone-splitting cavity (Figures and ), and covered by two pieces of PRF membrane and the resorbable membrane (Haiao Heal-ALL, ZH-BIO) (Figures and ). Surgical sites were closed with tight suture, and patient was instructed to take clindamycin and ornidazole for 7 days against infection.
After 6 months, CBCT revealed a horizontal augmentation of 2-3 mm in the alveolar ridge (Figure ), and conventional dental implantation (#35:3.3 mm(ф) × 8 mm(L), #36:4.1 mm(ф) × 8 mm(L), #37:4.1 mm(ф) × 8 mm(L); Straumann®, Switzerland) (Figure ) was performed under local anesthesia. At 3 months after implant placement, all-ceramic crowns were made to finish the final prosthodontics (Figures and ).
A significant bone increment was obtained at the horizontal direction of implant site after the application of modified ultrasonic osteotomy technique. The average bone augmentation at the horizontal direction was approximately 2-3 mm. The final implant prosthodontics was achieved with a solid and stable bone quantity without obvious bone resorption for 2 years after implant placement (Figures and ).


Case 3 was a 42-year-old female patient without systemic diseases. With 35, 36, and 37 lost for severe dental caries for over 10 year, the residual alveolar crest for such sites were shown as “blade shaped” (Figures , , ).
Ultrasonic piezosuegery induced bone splitting with simultaneous GBR was designed for this case, and followed by delayed implant placement.
The same surgical procedure is performed at the site of surgery (35-37) (Figures , , ). After the bone splitting, a Bio-oss bone substitution was placed into the surgical area (Figures and ), and two pieces of PRF and biological membrane (Haiao Heal-ALL, ZH-BIO) were finally applied to cover the GBR region (Figures ,). Other treatments and postoperative instructions were the same as in case 1 and 2 (Figure ).
After 6 months, CBCT images indicated an average augmentation of 2-3 mm in the width of residual alveolar ridge width (Figure ). Under local anesthesia, a minimally invasive dental implantation technique was performed by nonflap implant placement, three Straumann implants(#35:4.1 mm(ф) × 10 mm(L), #36:4.1 mm(ф) × 10 mm(L), #37:4.1 mm(ф) × 8 mm(L); Straumann®) were implanted in the augment alveolar ridge of 35 and 36 (Figures and ). The X-ray examination at 3 months after surgery indicated an ideally osseointegration and sufficient bone quantity at the implant-bone interface (Figures and ).
The ultrasonic piezosurgery induced bone-splitting technique dramatically the bone quantity at edentulous region, the horizontal bone quantity was augmented by 2-3 mm on average (Figures and ).


A 14-year-old girl, living in a refugee camp, was referred from a district hospital for the management of altered level of consciousness, fever, and hyperglycemia. Fever started 2 weeks prior to consultation for which she received empiric treatment for malaria in a district hospital. She was also diagnosed with severe acute on chronic malnutrition for which she received ready-to-use therapeutic food. The situation was worsened by polyuria, excessive thirst, uncontrolled hyperglycemia, persistent fever and progressive lethargy, necessitating her transfer to the endocrinology unit of the Mother and Child Center of Yaounde.
Past history was remarkable for diabetes which was diagnosed a year ago after 2 months of polyuria and excessive thirst. Insulin therapy was started at 0.5 IU/Kg/day with mixed insulin but patient's compliance was poor. She reported loss of sight 7 months ago (3 months after the diagnosis of diabetes). She is the 3rd child in a nonconsanguineous family of 10 children of whom three are alive. They fled the Central African Republic to Cameroon and for the past 4 years have been living in a refugee camp in the East region of Cameroon. Neither she nor the mother went to school.
On admission, she was lethargic (Glasgow coma scale was 13/15). Her weight was 19 kg (<3rd percentile), her height was 133 cm (<3rd percentile) for a BMI of 10.7 kg/m2, −5.5 SD for age. Vital signs were as follows: blood pressure: 100/80 mm Hg, pulse: 100 bpm, respiratory rate 34 cycles/min and temperature was 36.8°C. She had fine hair and poor dental hygiene. Her Tanner stage was B1P1. Her abdomen was distended with no palpable mass. She had vulvovaginal erythema. She had a dry mouth, sunken eyes, mixed signs of dehydration and severe malnutrition (skin pinch went back slowly) and bilateral leucocoria.
Her serum glucose level was 440 mg/dL (24.4 mmol/L), K + 3.3 mmol/L, corrected Na + 148.4 mmol/L, Cl-108 mmol/L. HbA1C was 12% (108 mmol/mol). Urinalysis revealed nitrites (3+), ketones (2+), glycosuria (3+), and culture later on showed growth of E coli. Her hemoglobin level was 12.3 g/dL, white blood cell count was 9500/mm3, and platelets count was 308 000/mm3. Aspartate aminotransferase (ASAT) and alanine aminotransferase (ALAT) were in the upper limits (42 and 44 IU/L, respectively) and renal function was normal (blood urea nitrogen (BUN) 0.07 g/L, serum creatinine 3 mg/L). Blood proteins were 93 g/L. Blood gases were not available.
She was admitted to the intensive care unit. She received oral rehydration solution for the malnourished (10 mL/kg/hr for 2 hours) followed by 1.5 L/m2/day of normal saline intravenously (IV) and KCL 1.5 g/L (IV) for 24 hours. Rapid-acting insulin was started 2 hours later with an hourly subcutaneous dose of 0.1 IU/Kg. An antibiotic was also given (ceftriaxone at 50 mg/kg/day) as well as routine de-worming with a single dose of 400 mg of albendazole. Four hours following admission, she was more reactive; ketonuria was 1+, serum glucose level was 408 mg/dL (22.4 mmol/L). Rapid-acting insulin was replaced by multiple injections using neutral protamine Hagedorn insulin (NPH) and actrapid (1IU/kg/day). Ready-to-use therapeutic food F75 (75 kcal/100 mL) was started at 130 mL/kg/day divided into eight meals. Folic acid (5 mg once) and vitamin A (200 mg on days 1, 2, and 14) were added to the treatment.
The day after admission, she developed lower limb edema and abdominal distension without ascites and IV fluids were stopped. The oedema regressed on day 2. On day 3, the F75 was replaced with F100 (100 kcal/100 mL) 130 ml/kg/day and progressive introduction of Plumpy Nut® and normal food. Progression of her nutritional status is shown in Table .
Communication with and education of patient and her family was difficult because of unavailability of a translator trained in medical terminology and the fact that the patient was almost blind. This was partially improved following her cataract surgery performed 2 months later.
Upon discharge after 9 weeks spent in hospital, her weight was 29 kg, height 133 cm (BMI 16.4 kg/m2) and MUAC of 160 mm. She was discharged to her refugee camp on multiple insulin injections. Two months later, her weight was 32 kg, height 135 mm (BMI 17.56 kg/m2) and HbA1 C 10%.


An 82-year-old Australian female was referred by her general practitioner to a general surgeon for biopsy +/- excision of a clinically up to 15 mm lesion of the mid-nose, which had been rapidly enlarging in size over the previous two to three weeks. The lesion was subsequently managed surgically by wide local excision and closed with an advancement flap. The histopathology report described an ulcerated tumor composed of undifferentiated small cells invading into the superficial subcutis. The cells stained strongly positive for cytokeratin-20 (CK-20), chromogranin, synaptophysin, and CAM 5.2; and stained negative for both thyroid transcription factor-1 (TTF-1) and CK-7. The immunohistochemical profile was therefore consistent with diagnosis of MCC []. In terms of the surgical margins, the peripheral margins were clear by at least 3 mm and the deep margin was seen to be clear by approximately 1.5 mm. There was neither lymphovascular space invasion nor perineural invasion seen.
The case was subsequently discussed at the hospital’s multidisciplinary (MDT) specialities meeting, with the consensus recommendation for discussion of postoperative radiotherapy under radiation oncology. Review of staging CT imaging three weeks postexcision showed features concerning for local recurrence just adjacent to the surgical scar site on the left side of the nasal bridge, or left nasal region of the maxilla. On review in the radiation oncology clinic, the suspicious region had a palpable nodule underneath it with firm and hard consistency, felt to be just less than 5 mm in diameter. Fluorodeoxyglucose-18 positron emission tomography (F-18 FDG PET) staging scan was subsequently organized in addition to a biopsy from this suspicious nodule.
The PET scan confirmed the clinical findings of loco-regional recurrence postsurgery, with a moderately glucose avid >10 mm (in short axis) subcutaneous nodule over the left anterior maxilla adjacent to the nose (as described clinically), an avid >10 mm (in short axis) enlarged left Level IB node just anterior to the left submandibular gland, and an equivocal <10 mm (in short axis) right Level IB node adjacent to the submandibular gland.
Fine needle aspiration (FNA) of the left cheek lesion showed specimen demonstrating paranuclear dot positivity for CK-20 and positivity for synaptophysin and chromogranin, and CAM5.2; TTF-1 and CK7 immunohistochemistry were again negative, consistent with MCC [].
The case was then further discussed at a large metropolitan centre Head and Neck MDT. By that time, the recurrent nodule on the left nasal region of the maxilla had grown to up to 3 cm with overlying erythema, and on examination there was palpable Level II cervical nodal regions bilaterally (Figure ). The MCC was therefore now consistent with T1N2M0, Stage III disease []. The consensus recommendation from the MDT panel was for definitive intent radiotherapy to the nose, to the bilateral neck, and to consider the bilateral cheeks. The patient’s ECOG status was 1.
The patient was treated to a total dose fractionation of 60 Gray in 30 fractions, 2 Gray per fraction, daily for six weeks, with 6 megavoltage (MV) photons, using volumetric modulated arc therapy (VMAT). A simultaneous integrated boost (SIB) technique was employed, in which the left nasal region of the maxilla and bilateral Level IB cervical nodes received 60 Gray in 30 fractions, and a total dose of 50 Gray was administered to the bilateral facial nodes, resected nasal disease (with a margin), bilateral parotids, and intervening lymphatics. A 1 cm custom tissue equivalent material (commonly referred to as bolus) was applied over the left nasal region. A mouthbite was used, and the nose was packed with gauze. A gross tumour volume (GTV) representing the recurrent nodule on the left nasal region of the maxilla was outlined; there were further expansions with a clinical tumour volume (CTV) expansion of up to approximately 5 mm and a planning target volume (PTV) expansion of up to 10 mm for the structures treated to 50 and 60 Gray as described above (Figures -).
The patient was also referred for a discussion of concurrent carboplatin chemotherapy under medical oncology []. Carboplatin was poorly tolerated, leading to a hospital admission, and chemotherapy was subsequently withdrawn after a single cycle. At that point, there was already noted in the clinic to be a marked reduction in the size of the lesion with radiotherapy. By the completion of radiotherapy, there was essentially complete remission clinically of the lesions of both the left nasal region of the maxilla and the bilateral Level II cervical nodes.
Both parotids were included in the treatment field. The mean dose to the bilateral parotid glands was between 53 and 54 Gray. Approximately 23% of the total volume of the mandible received a dose of 50 Gray, with no ‘hot spots’ (as defined by ICRU50 as a volume outside the PTV receiving dose greater than 100% of the specified PTV dose) in the mandible. Acute toxicities [defined by Common Terminology Criteria for Adverse Events (CTCAE) Version 4 as occurring within 90 days of treatment] reported by the patient included Grade I xerostomia with reduced saliva without significant dietary alteration, Grade I dermatitis with mild erythema in the treatment field, Grade I fatigue, and Grade I facial oedema with localized edema. Late toxicities, defined by CTCAE as occurring more than three months after the completion of radiotherapy, included the persisting or consequential Grade I xerostomia; and Grade I facial edema with localized edema. The relatively mild reported xerostomia by the patient both in the acute and late stages was anomalous given that there was no sparing of either parotid gland from the target volumes. However, approximately 18 months postcompletion of radiotherapy, a lower right premolar tooth was extracted by the patient’s local dentist. The cause was thought by the treating dentist to be in the context of xerostomia and dose received to the mandible.
The PET to assess treatment response three months postcompletion of radiotherapy demonstrated a complete metabolic response. All subsequent imaging have demonstrated complete response with no evidence of recurrence or distant disease. This has correlated with findings on clinical examination on three-six monthly reviews (Figure ). The patient remains well and has 6-12 monthly interval CT re-staging.


A 71-year-old female with a pertinent history of essential hypertension, anxiety, and hypothyroidism presented with acute-onset abdominal pain. The pain described the typical characteristics of pancreatitis, with sharp mid-gastric pain radiating towards the back, without any significant relieving factors, accompanied by unremitting nausea and vomiting. The patient did not have any recent history of smoking, drinking, or any recreational drug use. Her home medications included losartan, Synthroid, Xanax, and bupropion.
Clinical history did not yield any relevant information, apart from the fact that the patient had presented with similar acute symptoms of pancreatitis approximately four weeks prior to this episode. She was conservatively managed with intravenous fluids and pain control with the resolution of symptoms. The patient was investigated for common causes of pancreatitis, including ultrasound and computed tomography (CT) of the abdomen, lipid profile, drug screen, and hepatitis panel. The workup to rule out the common causes of pancreatitis was negative.
The patient subsequently improved and was discharged home to be followed by the gastroenterology for further investigations, including magnetic resonance imaging (MRI) of the abdomen and immunoglobulin (IgG4) levels, to rule out uncommon causes of pancreatitis. Of note, before discharge home, she was resumed on losartan at the same dose.
In this admission, the patient presented with similar complains and the repeat CT abdomen with intravenous (IV) contrast redemonstrated acute pancreatic inflammation with mild peri-pancreatic fluid accumulation without any evidence of necrosis and ductal dilatation (Figure ). The ultrasound of the abdomen was also repeated, which did not yield any evidence of cholelithiasis and the common bile duct measured 4 mm in size. The patient was evaluated by gastroenterology and was managed symptomatically. MRI the abdomen did not demonstrate intra or extrahepatic biliary ductal pathology, cholelithiasis, or choledocholithiasis. No evidence of abnormal pancreatic ductal pathology was noted. The patient was ultimately taken off losartan and started on calcium channel blocker for hypertension. The patient ultimately improved and was discharged home to do a follow-up with her primary care physician and gastroenterology as needed.
The patient had no evidence of abnormal biliary tract or pancreatic duct pathology on relevant imaging studies. Repeat lipid profile, hepatitis panel and liver function tests were within normal limits. The patient did not have alcohol use disorder, and blood alcohol levels were also negative on both admissions. Losartan was deemed as the causative agent for recurrent pancreatitis, a rare phenomenon to be described in the medical literature.


The patient was a 62-year-old female of African American ethnicity, with a past medical history of type 2 diabetes mellitus, chronic kidney disease secondary to diabetes, glaucoma, macrocytic anemia, chronic back pain and hypertension, who initially presented to her ophthalmologist with a small growth over her left upper eyelid. The patient had the swelling for more than four years that had gradually increased in size. She did not have it evaluated as it very occasionally caused symptoms like on and off tearing, itching, and irritation. She decided to seek medical care when she noticed some tenderness over the lesion. On initial evaluation, she was noted to have a 0.6 mm, elevated, cystic-appearing subcutaneous nodule in the central medial left upper lid margin and lash row. It was noninflamed and tender to palpation. The lesion was in close proximity to the cornea. All other eyelid and orbital functions were normal.
The lesion was initially thought to be a benign cyst. As it was symptomatic, it was excised and reconstruction was done. Histopathological analysis revealed positivity for carcinoma cells and suggested that it was a mucinous colloid carcinoma. As the margins were positive, a repeat excision with wider margins and reconstruction via advancement of her lateral full thickness upper lip into the central upper lid was done following a superior cantholysis of the lateral canthal tendon. The surgeons felt that there was no evidence of regional extension beyond what was excised. This was further confirmed by CT imaging of the orbits that revealed no locoregional extension or residual tumor (Figure ). Histopathological exam showed that the tumor was present within the dermis abutting the orbicularis muscle and was composed of lobules of epithelial cells floating in pools of mucin. Small ductal structures were observed within the lobules. A dual population of epithelial cells was identified with mild to moderate pleomorphism admixed with some atypical mitotic figures. Adjacent to the tumor, there were distended ductal structures with atypical proliferation of epithelial cells with a cribriform architecture and extracellular mucin. The tumor extended into the deep tissue edges, but perineural invasion was not identified. Immunohistochemical analysis revealed that the tumor was positive for tumor protein p63 (P63) and cytokeratin 7 (CK7) and negative for cytokeratin 20 (CK20), thyroid transcription factor-1 (TTF-1), and human epidermal growth factor receptor 2 (HER-2). These findings confirmed the diagnosis of PMA of the eyelid.
Given that an underlying occult malignancy of the gastrointestinal tract or breast may present similarly, she was evaluated extensively with CT scans of the thorax, abdomen, and soft tissues of the neck. She also had a mammogram and a colonoscopy. All the tests were unremarkable and negative for malignancy. Given the rarity of the disease, Radiation Oncology and Medical Oncology services were integrated to her care. She is being followed up in regular intervals and has not had any recurrence of her tumor thus far.


The patient was an 89-year-old woman. She had educational background with high school graduation. She had no medical history except for hypertension, and no family history of neuropsychiatric illness. She had helped for her son’s home business; however, she gradually seemed to lose her energy with hearing loss and muscle weakness. She began to complain her oral cenesthopathy, which is a feeling of filament structures in her mouth, and visited another hospital at the age of 88. She developed progressive cognitive decline with decreasing Mini-Mental State Examination (MMSE) score from 26 to 22 in half of a year, and become depressive. She was treated with mirtazapine up to 22.5 mg/day for her depressive symptoms; however, her complaint had changed to delusional parasitosis, which is a belief of filaria infection in her nose and eyes, and her parkinsonism, such as resting tremor, muscle rigidity and bradykinesia, had developed. She admitted to our department for the purpose of examination and treatment for her cognitive impairment, delusion and parkinsonism. Her MMSE score was 22, which lost some points on orientation (− 1), serial-7 (− 3), three-stage command (− 1), delayed recall (− 2), and copy of double pentagon (− 1). The score of Geriatric depression scale was 8, and that of the United Parkinson’s Disease Rating Scale part 3 was 45. The results of blood test were normal, and an electroencephalogram was unremarkable. In her brain images, a brain magnetic resonance imaging scan showed mild atrophy of prefrontal cortex and bilateral hippocampus, and a single photon emission computed tomography showed reducing cerebral blood flow in bilateral thalamus, in addition to the parts of bilateral prefrontal, temporal, occipital lobe and parietal association area. Furthermore, 123iodine-metaidobenzylguandine myocardial uptake was reduced. Considering all the results of examinations, she was diagnosed as probable DLB. She was treated with donepezil up to 10 mg/day for a month; however, she continued to complain “Insects and snakes enter my body from my anus and deprive my nutrition”. For her depressive symptoms, aripiprazole augmentation with mirtazapine was performed, and her parasitosis delusion was disappeared keeping dose of mirtazapine 22.5 mg/day and aripiprazole 3 mg/day for a month. After that, she developed aspiration pneumonia, and her delusional parasitosis was exacerbated along with discontinuation of aripiprazole and mirtazapine. Finally, she was treated with a combination of donepezil 5 mg/day and aripiprazole 1.5 mg/day for a month, and her parasite delusion was disappeared.


A 64-year-old man presented to our outpatient clinic with complaints of general fatigue and high fever (>38°C) at night that persisted for 3 days. He had previously undergone 25-mm mechanical aortic valve implantation (St. Jude Medical Regent; St. Jude Medical, Inc., St. Paul, MN) 12 years prior because of severe aortic regurgitation. Upon examination, no focus of infection could be identified, and he was admitted to our heart centre to exclude the possibility of PVE. After three sets of blood culture were obtained, intravenous ceftriaxone (2 g/day) was empirically initiated. Initial transthoracic echocardiography (TTE) revealed no dysfunction of the mechanical aortic prosthesis, and whole-body CT revealed no embolic events. The patient recovered to a low-grade fever (37–38°C) 48 h after the initial antibiotics therapy and showed improved C-reactive protein level (from 13 to 6.3 mg/dL) in the first 5 days. Subsequently, transoesophageal echocardiography (TOE) performed on the 5th day of hospitalization revealed a 6–11 mm mobile echogenic mass attached to the prosthetic valve and a thickened area within the posterior annular region with a low echogenic appearance at the level of the non-coronary sinus of Valsalva (). At this point, the patient met two minor diagnostic criteria (predisposing heart condition of prior prosthetic valve implantation and high fever) in the modified Duke diagnostic criteria for infective endocarditis because the TOE findings were considered inconclusive. Subsequently, combined 18F-FDG PET/CT and CT coronary angiography revealed intense hyper-metabolism above the aortic valve prosthesis with greater intensity at the posterior end and absence of obstructive coronary artery, thereby confirming the diagnosis of aortic PVE complicated with perivalvular abscess (). Following a comprehensive discussion with the endocarditis team, including an infectious disease specialist, a microbiologist (pathologist) and cardiovascular surgeons, we decided to switch from intravenous ceftriaxone to appropriate antibiotic therapy [intravenous ampicillin (8 g/day) and gentamicin (180 mg/day)] for blood culture-negative patients with PVE based on the current ESC guideline and withheld specific serological tests or polymerase chain reaction (PCR) assays for unusual but potential microorganisms as long as the patient’s clinical course was not out of track and the antibiotics therapy appeared clinically effective. Following 1 week of intensive antibiotic treatment, we finally decided to perform surgery mainly because the PVE was complicated with perivalvular abscess, as evidenced by 18F-FDG PET/CT with all negative blood culture tests. On the 14th day of hospitalization, prosthetic valve was surgically replaced with a 25-mm bioprosthesis (25A Trifecta™; St. Jude Medical, Inc., St. Paul, MN, USA) with extensive debridement of the infected and necrotic tissues surrounding the aortic annulus (). Surgically excised tissue was subjected to microbiological and pathological analysis; however, no evidence of bacterial organisms was found () and blood culture tests were negative. Post-operative TTE revealed a normally functioning aortic valve, and he was discharged on the 51st day. Based on the international guideline, antibiotic therapy was continued for 6 weeks until hospital discharge, although gentamicin was discontinued in the first 3 weeks because of nephrotoxicity. Furthermore, intravenous antibiotic treatment was followed by 6-month outpatient oral antibiotic therapy with amoxicillin (750 mg/day) without any recurrences.


A 90-year-old male with a past medical history of hypertension, chronic kidney disease stage II, and hyperlipidemia presented with complaints of intermittent hematuria. He had no prior history of mucosal bleeds and denied having any trauma. He denied having any history of easy bruisability. He had no pain. He has no prior history of hematuria and did not have any prostate issues. He had a history of cholecystectomy and left hip replacement. He had no current or past history of smoking, illicit drug use, or alcohol use. He had no history of taking any herbal or traditional medications. He did not have any significant medical issues in his family and family history was negative for any cancers or bleeding disorders. His temperature was 98.6° F, blood pressure 134/87 mmHg, pulse 83/minute, and respirations 14/minute. Physical examination was unremarkable for any acute findings.
Initial workup revealed hemoglobin (Hb) of 8.9 g/dl, hematocrit (Hct) of 27.1%, white blood cell count (WBC) of 9.4 10*3/uL, and platelet count of 235 10*3/uL. The metabolic panel was unremarkable and revealed electrolytes and liver function tests within the normal range. The patient's BUN and creatinine were 58 mg/dl and 1.3 mg/dl respectively which were also at baseline for him. His activated partial thromboplastin time (aPTT) was found to be mildly prolonged at 48.4 seconds. But prothrombin time (PT) was 11 seconds and international normalized ratio (INR) of 1.1, both within normal limits. The prostate-specific antigen was checked and came back at 1.2 ng/ml. Urinalysis was negative for nitrites, leukocyte esterase, and bacteria and showed only 0-1 white blood cells but showed a large amount of blood with more than 100 red blood cells. Peripheral smear was done which showed normocytic, normochromic anemia with mild anisocytosis. White blood cells and platelets showed no abnormality (Table ).
A chest X-ray was done as a part of the routine investigations and returned normal (Figure ).
The patient was admitted with urology consultation and underwent a cystoscopy where no active bleeding was found and a small clot in the urinary bladder was evacuated (Figure ). The patient then stopped bleeding for 24 hours but then again started having hematuria again which was more severe this time compared to the time of admission. Also, he started bleeding from the right arm where he had an intravenous line that had been placed earlier.
At that time, Hematology consultation was obtained; aPTT was repeated which was mildly prolonged at 44.8 seconds. Factor VIII and Factor IX assays were performed and showed results of less than 3% (<3%) and 27% respectively. Factor VIII inhibitor levels were 12 BU/ml per Bethesda assay. His Hb was repeated which came back at 7.0. He was transfused with two units of blood.
During this admission, he was started on corticosteroids with methylprednisolone being given intravenously initially at 80 mg followed by a prednisone tablet at 40 mg. The patient was also started on folic acid 1 mg and ferrous sulfate at 325 mg. The patient was simultaneously started on recombinant porcine factor VIII intravenously with 200 units/kg. His hematuria and bleeding from the right arm subsided within 24 hours. Factor VIII assay came back at 258%.
He was discharged to an inpatient rehabilitation unit. However, after five days in rehabilitation, he had a recurrence of hematuria. Factor VIII assay was done again and values came back at 140%. This was followed by a recurrence of bleeding from the right arm on the next day. At this time, the patient was readmitted to the medical floor from the rehabilitation unit. Factor VIII assay was done again and came back with a value of 9%. Factor VIII inhibitor levels were checked again and returned at 12 BU/ml per Bethesda assay again.
The attempts to rid the patient of neutralizing antibodies to factor VIII with corticosteroids were only partially and temporarily successful. Unfortunately, even after a full recovery initially with recombinant porcine factor VIII, hematuria recurred and factor VIII levels were down again to 9%. This suggested that anti-factor VIII antibodies were now cross-reacting with recombinant porcine factor VIII. The patient was then started on recombinant factor VIIa as a bypassing agent at 90 mcg/kg and was repeated every three hours. Hemostasis was achieved at 24 hours and the patient did not have any further recurrence of hematuria after 48 hours after which he was discharged home with a follow-up appointment with Hematology as an outpatient. The patient was later on readmitted with pneumonia after two months but did not have any bleeding event at that time and his aPTT was 36.3. At a six-month follow-up, he had no more bleeding event and remained stable with aPTT of 35.9.


A 61-year-old man was transferred to the authors’ institution from another facility for the management of a suspected bronchoperitoneal fistula following an exploratory laparotomy to address a late complication from a prior segmental colectomy. The patient’s past medical history was significant for a near-obstructing sigmoid colon adenocarcinoma. This was treated with a laparoscopic converted to open-extended left hemicolectomy with colo-rectal anastomosis. His immediate post-operative course was unremarkable. He presented to the outside hospital three months after this index operation reporting abdominal pain and early satiety. Computed tomography (CT) imaging showed a large, walled-off collection of fluid and debris in the left upper abdominal quadrant (Figure ).
The collection appeared to abut the left diaphragm and extended inferiorly and across the midline (Figure ).
The patient subsequently underwent an exploratory laparotomy, evacuation of a feculent phlegmon, completion colectomy, and ileorectal anastomosis with diverting loop ileostomy creation. There was an inadvertent gastrostomy and two duodenostomies which were repaired primarily and buttressed with jejunal serosal patches. On post-operative day 2, the patient developed acute hypoxic respiratory failure requiring intubation and mechanical ventilation. On examination, air was heard escaping from the patient’s abdominal incision with every breath. The patient was taken for a CT scan of the chest, abdomen, and pelvis to assess for pneumonia or abdominal sepsis as a source of his deterioration. It demonstrated a left bronchoperitoneal fistula (Figure ).
The patient was then transferred to the authors’ institution for higher level of care and input from cardiothoracic surgeons. After he was resuscitated in the intensive care unit, the patient was taken back to the operating room to evaluate and control the bronchoperitoneal fistula. Upon entering the peritoneal cavity through the prior laparotomy incision, air was heard escaping into the abdomen coinciding with the patient’s ventilated breaths. The abdomen was irrigated with saline, and air bubbles were seen percolating through the left chest into the abdomen. The prior duodenostomies were re-buttressed with jejunal serosal patches. A feeding catheter jejunostomy (FCJ) was placed for enteral feeding access. Two large drains were placed in the left subdiaphragmatic space to control the fistula. Primary repair of the diaphragm defect was not attempted due to significant inflammation and immobility of the stomach. The patient was returned to the intensive care unit in stable condition.
The abdominal drains were connected to a Pleur-evac chest drainage unit and placed to wall suction. An air leak was noted, but it resolved by post-operative day 5 despite continuous positive pressure ventilation. The drains were disconnected from suction approximately two weeks post-operatively. However, several days later air was noted escaping from the abdominal incision. Therefore, the drains were placed back to suction, and evidence of pneumoperitoneum was resolved.
Approximately one month into his hospital stay, the patient developed an acute pulmonary embolus and was initiated on therapeutic anticoagulation. He subsequently experienced an intra-abdominal hemorrhage. The abdomen was re-explored in the operating room, but no bleeding source was identified. At this time, there was no audible air passing into the abdomen from the left chest. However, two enterocutaneous fistulas were noted, one originating from the gastroduodenal junction and the other involving the jejunum. Additionally, one of the patient’s prior duodenotomy repairs had dehisced. Given that this duodenal injury failed to heal after two earlier repairs, a robnel drain was placed retrograde into the duodenum through this duodenotomy. Malecot drains were placed in each fistula and externalized to serve as one controlled fistula. A large ostomy appliance was fashioned to encompass much of the patient’s abdomen. The fistulas were managed with maximum medical therapy including nil per os (NPO) status and total parenteral nutrition (TPN).The patient spent 91 days in the hospital. He experienced multiple medical complications, including respiratory failure requiring tracheostomy, fungemia, and malnutrition. He was discharged to home with home health services to assist with TPN infusions.
The patient’s nutritional status was optimized while on TPN, and fistulous output was managed with anti-secretory medications. Despite these interventions, the fistulas continued to drain approximately two liters of fluid per day. He required frequent intravenous fluid replacements, and his quality of life was greatly affected. Eventually, the patient was offered a fistula takedown.
Approximately one year after discharge from the hospital, the patient returned for enterocutaneous fistula takedown, FCJ placement, and complex abdominal wall reconstruction. At this point, we had determined the patient had one gastroduodenal fistula as well as a jejunal fistula. The enterocutaneous fistulas on the day of the operation are shown in Figure . We performed a primary closure transversely of the gastroduodenal fistula in a Heineke-Mikulicz pyloroplasty fashion. The jejunal fistula was resected. We left the loop ileostomy in place to avoid a second anastomosis. Bilateral separation of components was necessary to close the abdomen. We also placed an absorbable mesh in an overlay fashion.
The patient’s post-operative course was notable for candidemia and bacteremia, likely secondary to his peripherally inserted central catheter which was used for home TPN infusions. Infectious disease specialists were consulted, and the patient completed appropriate antimicrobial therapy. He was discharged to home after 26 days in the hospital tolerating a normal diet and nocturnal feeds through the FCJ. At his two-week follow up-appointment, the patient demonstrated adequate weight gain, and the FCJ was removed.


An 11-year-old boy presented with a several-month history of left knee pain. After osteosarcoma was confirmed from an incisional biopsy specimen, we planned a limb salvage operation with tumor prosthesis. Although limb salvage operations with tumor prostheses usually require ABTs, the patient and his family did not want ABTs due to their religious beliefs, despite the patient's anemic conditions caused by neoadjuvant chemotherapy. Two weeks before surgery, the boy's Hb level was 7.5 g/dL. Following the previously described protocol, we administered rHuEPO and iron supplements, which increased his Hb levels to 11.0 g/dL by preoperative day 2. Surgery involved amputation or limb salvage operation with biologic reconstruction or tumor prosthesis (Fig. ). After hypotension was induced, 1 g of tranexamic acid was diluted to 20 mL and continuously administered (4 mL/h) during the operation. The total operative time was 4.5 hours, with approximately 450 cm3 of intraoperative blood loss; a postoperative total hemovac drain check revealed a total collected volume of 550 cm3. By postoperative day 3, the patient's Hb dropped to its lowest level (6.6 g/dL). However, his laboratory findings gradually improved while following our protocol, with no anemia symptoms such as dyspnea or tachycardia (Fig. ). The patient was maintained on the protocol for 2 weeks postoperatively until his Hb level recovered to 11.2 g/dL.


A 69-year-old man presented to our dermatology department with a widespread rash consisting of multiple brown to erythematous, keratotic papules, with crusted centers, involving seborrheic areas such as scalp, ears, lateral aspects of the neck and chest (presternal area). Some lesions were scattered individually and others were confluated into larger, crusted plaques (Figure , ). The patient also presented flexural involvement, with maceration and large vegetative plaques in the genito-crural folds, associated with malodour (Figure ). The lesions were itchy and the patient complained about the disfigurement, pain and malodour of the intertriginous lesions. Physical examination also showed acral abnormalities and oral involvement. Skin-colored, wart-like, asymptomatic, flat-topped papules were noticed on the dorsa of the hands (Figure ). The nail changes were subtle with a few discrete white and red alternating longitudinal bands and notching of the distal nail plates (Figure ). Oral lesions consisting of white cobblestone papules with a central depression were observed on the buccal mucosa, bilaterally (Figure ).
The widespread rash developed two years ago, with no obvious identifiable trigger. The patient reported that starting his mid-twenties he noted the occurrence of small scattered papules limited to the face. Along the years he presented to several dermatology clinics and received treatment for acne or recurrent staphylococcal skin infection. The lesions improved with various topical treatments, but always recurred and in the ensuing two years, the eruption continued to progress in the seborrheic and flexural areas, worsening dramatically.
The clinical aspect was suggestive of Darier disease. The patient couldn’t provide a family medical history and no other member of his family was available for analysis.
Routine laboratory tests were within normal limits. A punch biopsy of a hyperpigmented keratotic papule on the patient’s trunk showed focal acantholytic dyskeratosis - “corps ronds” (round, large, acantholytic keratinocytes predominantly located in the stratum spinosum and stratum granulosum) and “grains” (elongated, small, acantholytic cells, mostly located in the stratum corneum) (Figure ). Bacterial and fungal cultures of samples from the genito-crural folds were positive for yeast (Candida albicans), as well as for Pseudomonas aeruginosa and methicillin-sensitive Staphylococcus aureus.
Based on clinical and histopathological grounds, the diagnosis of DD with superinfected lesions was rendered. Intravenous antibiotic therapy with piperacillin plus tazobactam and ciprofloxacin was administered within the 10 days of hospitalization. Treatment with oral isotretinoin 20 mg per day was also initiated. The topical treatment consisted of antiseptic washes with chlorhexidine gluconate, fusidic acid 2% cream, ketoconazole 2% cream and emollients. After two months, at follow-up, the results were extremely satisfactory (Figure ).


A 60-year-old female is described who presented with atrial premature beats and paroxysmal atrial fibrillation. A transesophageal echocardiogram (TEE) was performed which showed a small, localized thickening of the interatrial septum that was considered as a structural variant (Fig. a). She subsequently underwent circumferential pulmonary vein radiofrequency ablation (RFCA). Three months after RFCA, the patient was readmitted following the sudden onset of chest tightness, dyspnea, transient palpitation, and recurrent pericardial effusion. Chest x-ray examination revealed moderate cardiomegaly and T-wave abnormalities in precordial leads were found in electrocardiogram. Transthoracic echocardiography (TTE) examination revealed a large immobile, non-pedunculated mass (47 mm × 17 mm × 11 mm) occupying almost half of the right atrium (RA) (Fig. b) and extending along the lateral wall of the RA to the interatrial septum. The mass did not extend into the superior vena cava (SVC) and did not cause stenosis of the tricuspid valve annulus. Transesophageal echocardiogram (TEE) confirmed the presence of the mass and showed that it extended from the orifice of SVC to the tricuspid valve annulus and from the right auricle (RAA) to the RA (Fig. c). The mass exhibited central cavitation suggesting that blood flow was poor in this region. No malignant or atypical cells were found in the bloody pericardial effusion.
Based upon these findings, our attention focused on the identity of the mass: cardiac tumor or hematoma? If it was a tumor, was it benign or malignant? If it was malignant, had it already metastasized? A F18-fluorodeoxy glucose-positron emission tomography (18F-FDG PET-CT) scan was advised to further characterization of the mass and assessment of metabolic activity. The scan revealed an area of abnormal tracer activity in a hard tissue mass occupying the RA and encroaching on the adjacent left atrial roof (Fig. a, b). With contour and heterogeneous contrast enhancement, the infiltrative mass was observed to arise from the RA. Moreover, another area of increased radiotracer activity was found in the RAA but there were no hypermetabolic lesions in adjacent organs. These images suggested the possibility of an angiosarcoma without metastasis. Subsequently, the patient underwent surgical excision of the tumor. Pathological analysis identified the tumor was a primary cardiac angiosarcoma. Microscopic findings showed that the tumor was composed of frequent slit-like vascular channels and hyperchromatic, elongated cells with obvious nuclear atypia (Fig. a). The results of immunohistochemical investigation revealed that tumor cells stained positive for CD31 (Fig. b), CD34, VI antigen, and vimentin which confirmed the endothelial origin of the tumor. Unfortunately, the patient refused treatment with adjuvant radiotherapy, chemotherapy or immunotherapy. Within 3 months post surgery, the tumor recurred in the same location. The patient survived for a total of 7 months from the first episode of tamponade.


A 48-year-old male was referred to the clinic for recurrent fever, cough and 5-kg weight loss during past 3 months. He denied breathing difficulties, chest pain, night sweats, wheezing and other uncomfortable symptoms. He was a smoker of 30 pack-years and had no history of travel or TB. There were no significant findings in his prior medical or family history.
At the time of the initial evaluation, the patient showed an auricular temperature of 39.2 °C, a blood pressure of 108/70 mmHg, a pulse of 108, and a respiratory rate of 20. He had diminished breath sounds on both sides, and no enlarged lymph nodes were noted. Examinations of the heart, abdomen, extremities and nervous system were normal.
Laboratory data showed that the patient’s white blood cell count, haemoglobin level and platelet count were normal. C-reactive protein and lactate dehydrogenase (LDH) levels were elevated at 151.00 mg/L (normal range: 0~8 mg/L) and 395.00 U/L (0~247 U/L). Tumour markers, such as CEA, NSE, SCCA, ProGRP and CYFRA21-1, were all in the normal range. HIV serology was also negative. Computed tomography (CT) of the chest showed bilateral cavitating lesions with mediastinal enlarged lymph nodes (Fig. , A-C). The patient was started on empiric antibiotic treatment with cefoperazone-sulbactam. After 3 days of therapy, his temperature was still above 38.5 °C. A CT-guided biopsy of the pulmonary cavity was performed on the 4th day after admission. Pathology revealed mild atypical alveolar epithelioid cells and chronic interstitial fibrous tissue proliferation with necrosis. The tissue was negative on smear and culture for acid-fast bacilli. Periodic Acid-Schiff (PAS) stain was also negative. The patient’s antibiotics were changed to imipenem-cilastin sodium and metronidazole. However, the second combination of antibiotics was ineffective. The patient was still febrile, and further blood cultures remained negative. On the fifth day, he underwent bronchoscopy and bronchoalveolar lavage, which were negative for any masses, abscesses or areas of bleeding. However, the patient tested positive for galactomannan antigenemia in bronchoalveolar lavage fluid (BALF) and blood. Aspergillus fumigatus complex was identified from the BALF culture on the 10th day. Due to the new microbiological findings, the patient was treated with voriconazole. In addition, the patient was still treated with linezolid, moxifloxacin and imipenem-cilastin sodium combined with metronidazole successively during the third round of antibiotic use. He was persistently febrile and developed breathing difficulties. We recommended the patient undergo positron emission tomography/computed tomography (PET-CT) to assess the possibility of haematological malignancies. It was refused because of the financial burden. We performed bone marrow aspiration on the 20th day. No abnormal cells were observed in the bone marrow examination. A CT scan performed 23rd day revealed further expanding consolidation and bilateral pleural effusion (Fig. , D-F). Thoracentesis was performed on the 24th day. Pleural fluid analysis revealed a red blood cell count of 16,320 cells/μL and a nucleated cell count of 1280 cells/μL (31% lymphocytes and 54% segmented cells), and the Rivalta test was negative. LDH and adenosine deaminase (ADA) levels were 381.00 U/L (0~247 U/L) and 15.00 U/L, respectively. There were no malignant cells in the pleural effusion. Considering the possibility of opportunistic pathogens, additional drugs were added to cover nocardia and pneumocystic infections. However, his temperature still increased to 40 °C. A second CT-guided biopsy was performed on 27th day to find the cause of repeated fever, and the pathological examination of the specimen revealed polygonal atypical lymphoid cell proliferation with necrosis. The second PAS staining and tissue culture were also negative. Immunohistochemical staining on 35th day showed positive markers for CD20, EBER, BCL-2, PAX5, MUM-1 and a Ki-67 rate of 70% (Fig. ), which was consistent with a diagnosis of diffuse large B-cell lymphoma (DLBCL). He also had a serum IgM test for Epstein–Barr virus (EBV) and other viruses, including influenza and toxoplasma, rubella virus, cytomegalovirus, herpes virus (TORCH), which were all negative. The EBV load was less than 5*103 (< 5*103). He was therefore diagnosed with synchronous pulmonary aspergillosis and DLBCL. Despite the combined R-COPE chemotherapy (rituximab 600 mg d 0 + cyclophosphamide 0.4 g d1–2 + vindesine 4 mg d1 + dexamethasone 20 mg d1–4 + VP-16 0.1 g d1–2) starting 38th days after hospitalization, the patient’s situation continued to deteriorate. He developed cervical, axillary and inguinal lymph node enlargement, which were unremarkable on admission. His LDH increased to 1627 U/L. The following CT scan (Fig. , G-I) showed bilateral pleural effusion, atelectasis and consolidations of all right lung lobes with air bronchograms. Then, the patient died of progressive respiratory failure on the 52nd day.


A 30-year-old woman was admitted to our hospital for dry cough lasting 1 month. She presented fever and chills, and her serum C-reactive protein levels elevated to 123.3 mg/L. Furthermore, her hemoglobin level was 8.9 g/dl, which was iron-deficiency anemia. Computed tomography (CT) revealed an approximately 6.4 cm × 2 cm-sized heterogenous soft tissue masses in both adnexa with splenomegaly, mass, and nodules of pericardial invasion of the right atrium with pericardial effusion and bilateral pulmonary edema. Thus, we performed positron emission tomography (PET), which revealed 2 adnexal masses with heterogeneous focal increased 2-deoxy-2-fluoro-D-glucose (FDG) uptake (maximum standardized uptake values (SUVmax 7.2) and heterogeneously increased FDG uptake along the pericardium (SUVmax 8.8), right pleura (SUVmax 4.3), bone marrows. Furthermore, splenomegaly with heterogenous increased FDG uptake (SUVmax 4.6) was observed (Fig. -A).
One week later, her leukocyte counts abruptly decreased to 2050/mm3; hemoglobin, 8.6 g/dl; platelet count, 181,000/mm3. Furthermore, a peripheral blood smear revealed a few immature cells (3%) (Fig. -A). Smears of bone marrow aspirates demonstrated increased medium to large sized blasts, including myeloblasts and monoblasts accounting for 32% of total nucleated cells and bone marrow biopsy revealed hypercellular marrow, with 90% cellularity (Fig. -B, C). The immunohistochemical profile of the tumor cells was as follows: positive for CD45 and myeloperoxidase (MPO) on granulocyte. Moreover, chromosomal analysis revealed a 46, XX, del18 (p11) [20] karyotype and cytogenetics examination, especially fluorescence in situ hybridization (FISH), including AML1/ETO rearrangement, PML/RARA rearrangement, CBFB/MYH11 rearrangement, MLL rearrangement, showed all negative results. The molecular markers, including a Nucleophosmin-1 (NPM1) gene mutation, CCAAT/enhancer-binding protein-a (CEBPA) gene mutation, Fms-like tyrosine kinase-3 gene(FLT3)-Internal tandem duplication (ITD) gene mutation were not detected in direct sequencing, and multiplex reverse transcriptase PCR assay (Hemavision, DNA technology, Aarhus, Denmark) showed negative result. The Hemavision panel include 28 different translocation or chromosomal rearrangements, including t(1;11)(q21;q23), t(8;21)(q22;q22), inv(16)(p13;q22), t(9,22)(q34;q11). Therefore, patient was finally diagnosed as acute myelomonocytic leukemia and she received remission-inducing chemotherapy comprising anthracycline and cytarabine, which is a standard regimen for AML. An anthracycline drug, such as daunorubicin or idarubicin, is given in a single intravenous (IV) dose on each of 3 days during the first week of treatment and cytarabine also given continuously for 7 days through an IV line. About 14 days after chemotherapy started, febrile neutropenia was developed and she received empirical antibiotics and granulocyte colony stimulating factor daily. After recovery from cytopenia, sequential bone marrow examination revealed no current residual leukemic cells. Furthermore, CT and PET follow-up examination revealed markedly decreased size and obliteration of FDG uptake of both ovarian masses, obliterated activity of heterogeneous FDG uptake along the pericardium and right pleural infiltration, along with decreased activity of splenomegaly and bone marrow (Fig. -B).
She received 1 cycle of post-remission chemotherapy with high dose of cytarabine twice daily at a 3 g/m2 dose on days 1, 3, and 5. Four weeks later, she underwent allogenic hematopoietic stem cell transplantation from Human leukocyte antigen (HLA)-identical sibling donor. Her bone marrow engrafted successfully without complications, and she visited our clinic regularly for a year with no evidence of leukemia relapse or graft-vs host disease.


In February 2016, an 81-year-old female visited our hospital due to left knee pain of increasing intensity. She presented the sudden onset of severe acute knee pain with long lasting knee pain for several years. On examination, this pain was localized to the medial aspect of the proximal tibia area. Mild swelling and joint effusion were evident; however, there was no sign of infection, such as heating sensation and redness. Laboratory studies, including blood coagulation test, fibrinolysis, and liver function tests, did not show abnormal findings.
The patient had suffered from bilateral OA of the knees for several years. She had been prescribed analgesics and nonsteroidal anti-inflammatory drugs at other primary clinics. Two months ago, she had undergone her 1st intra-articular corticosteroid (triamcinolone) injection of both knees. However, left knee pain was worsening and was localized to the medial aspect of the proximal tibia. At that time, plain radiograph of the knee obtained at other primary clinic showed a scalloping subchondral lesion of the medial femoral condyle of the left knee with mild joint-space narrowing (Fig. ).
We evaluated plain radiographs of the knee to identify the source of her left knee pain. Plain radiographs showed a well-demarcated radio-opaque lesion on the medial proximal tibial plateau was evident; this was not present on the radiographs taken 2 months prior (Fig. ).
We recommended total-knee arthroplasty (TKA) because the patient had received several conservative treatments, which did not relieve the pain and disability. We conducted a posterior stabilized TKA (Genesis II; Smith & Nephew, Memphis, TN), with no requirement for bone grafting or additional prosthesis, such as metal augments or stems. Pathologic examination of a specimen from the medial tibial plateau showed partial disruption of the articular cartilage and wedge-shaped ON. A higher-magnification image revealed bone necrosis with adjacent coagulative necrosis, and infarction of the fatty marrow relevant to ON. At the postoperative 1 year follow-up, the patient was satisfied with the surgery and had no pain during walking and active knee motion (Fig. A, B).


A 70-year-old male patient, nonsmoker, known to have hypertension, uncontrolled DM, Alzheimer's disease, benign prostatic hyperplasia and a history of recurrent urinary tract infections, presented to our emergency department with two days history of high grade fever and decreased level of consciousness. As reported by his family, he was complaining of abdominal distension and constipation since one week without any episode of vomiting. The patient has been recently treated for urinary tract infection with an unknown antibiotic that was intentionally stopped two days prior to his presentation to our department.
Upon arrival, vital signs showed a temperature of 39°C, heart rate of 98 beats per minute, blood pressure of 100/60 mmHg and a respiratory rate of 20 breaths per minute. The patient appeared obnubilated with a Glasgow Coma Scale (GCS) of 12. Physical examination was relevant for left-sided costovertebral angle tenderness, abdominal distension, and fecal impaction on digital rectal examination.
Initial laboratory tests were consistent with sepsis likely secondary to urinary tract infection with blood tests revealing a white blood cell count (WBC) count of 8,100/mm3 with 76% granulocytes, hemoglobin of 10.4 g/dl, platelet count of 248 000/mm3, creatinine level of 1.23 mg/dl, urea of 71 mg/dl, and CRP of 175. Urine analysis demonstrated 6 to 8 WBCs, 2 to 3 RBCs, positive nitrites, and numerous bacteria.
Urgent CT scan of the abdomen and pelvis revealed severe left hydronephrosis and hydroureter with the presence of gas in the left collecting system and left ureter (Figures and ). The aforementioned hydroureteronephrosis resulted from an extrinsic compression of the left pelvic ureter between an atherosclerotic external iliac artery and a giant fecaloma (). It is noteworthy that the ureter distal to the level of obstruction was not dilated ().
Urine and blood cultures were taken. Initial medical management consisted of intravenous fluids, broad-spectrum antibiotics (Meropenem and a single shot of Amikacin) and strict glycemic control. After failure of initial conservative measures of fecaloma evacuation by digital rectal examination and fleet enemas, an ultrasound and fluoroscopy-guided left percutaneous nephrostomy of 10 French (Fr) was inserted for drainage of the renal pelvis. Frank pus was aspirated, confirming a left pyonephrosis. An antegrade ureterogram was intentionally omitted in order to avoid any retrograde bacteremia in this septic patient.
A few hours following the nephrostomy insertion, the patient's confusion improved significantly. He became clinically and hemodynamically stable over the following days as the sepsis resolved. Oral laxatives and fleet enemas were given for fecaloma evacuation. Urine culture grew a multidrug-resistant Proteus Mirabilis. A follow-up noninjected CT scan was performed one week after the admission to document the resolution of the infection, the absence of renal abscesses and the resolution of the fecaloma (). An antegrade ureterogram was performed showing a patent ureter with no intrinsic lesions (); therefore the PCN was removed (in total nine days after patient's admission).


A 41-year-old pregnant woman, in her first gestation, sought medical care complaining of pain in her right shoulder for the last two months, associated with the impairment of moving her arm. She was 20 weeks pregnant and did not attend any prenatal consultation. Her medical history included smoking, correctly treated syphilis, and pulmonary thromboembolism. The patient did not have any family history of thyroid disease.
Right arm radiography () demonstrated a lytic bone lesion on her shoulder, apparently involving the surrounding soft tissues. The initial working diagnosis was a soft tissue or bone tumor with differential secondary lesions, for example, metastatic carcinomas. The patient was submitted to a biopsy of the lesion (), which revealed a high-grade malignant neoplasm with an epithelioid aspect on the histological examination. The predominant pattern was solid with sheets of atypical cells and a sparse microfollicular-like arrangement. There were many mitoses and, occasionally, nuclear hyperchromasia. The morphological hypothesis was a metastatic carcinoma and, more remotely, primitive neuroectodermal neoplasms.
The immunohistochemistry (IHC) was diffuse and strongly positive for cytokeratin (AE1/AE3) and TTF1. CD45, CD99, chromogranin-A, and synaptophysin were negative. Since the morphology was favorable for thyroid differentiation, the IHC panel was extended to thyroglobulin, calcitonin, and surfactant with a positive result only for thyroglobulin. Thus, the diagnosis of metastatic thyroid carcinoma was made. Also, the thoracic computerized tomography (CT) failed to show any pulmonary lesion. The thyroid examination revealed an increased cervical volume, with some firm and painful areas, in the paramedian region. It is important to mention that until that moment the patient had no previous medical care. The cervical enlargement observed in the anterior portion of the neck was not reported by the patient. When asked, the patient reported that there might have been some increase in the region.
The ultrasonography (US) and magnetic resonance imaging (MRI) revealed a suspected nodule localized in the left lobe of the thyroid, measuring 4.0 cm in diameter (). An FNA was performed, and cytological evaluation was consistent with category IV of Bethesda Classification, suspected for follicular neoplasm ().
After this diagnosis, the option for treatment was a total thyroidectomy. The macroscopic examination demonstrated a diffusely increased glandule where there was a nodule measuring 4.0 × 4.0 × 3.0 cm in the left thyroid lobe. It was an expansive tumor with macroscopic infiltration of the capsule ().
The histopathologic finding was similar to the bone/soft tissue lesion biopsy. Immunohistochemistry results were the same (). The final diagnosis was follicular carcinoma of the thyroid, with follicular (90% of the tumor) and solid (10% of the tumor) patterns accompanied by necrosis, vascular infiltration (at least six areas), and lymphatic infiltration. Mitosis number is two in ten high magnification microscopic fields. Near the thyroid, there were two lymph nodes without neoplasm. However, no specific resection of cervical lymph nodes was performed. The pathological stage was pT3a pN0 pM1 (AJCC, 2017). The tests performed were: IHC markers such as thyroglobulin and TTF1 with a diffuse pattern of immunostaining. The Ki-67 index was compatible with the mitosis number (8%), and the p53 marker was negative.
A cesarean was performed at the same time, at 30 weeks of pregnancy. After three months, the patient underwent surgery of the right arm to install a prosthetic. She received therapeutic iodine treatment and had medullary compression syndrome, secondary to the bone and spinal involvement. There were no other sites of metastasis. The disease progressed, and the patient died one year after the diagnosis. The child survived without morbidities.


A 62-year-old woman was diagnosed of thyroid cancer and received total thyroidectomy with left neck lymph nodes dissection (level II, III, IV, IV, and VI). The patient suffered left unilateral vocal cord immobility after the surgery, and she received type I thyroplasty with vocal cord medialization 1 month after the thyroid and neck dissection surgery as treatment for her phonatory insufficiency. The patient did not receive further chemotherapy or radiation therapy, and no evidence of residual thyroid cancer or recurrence had been noted in her follow ups. There was no complaint of swallowing difficulty and aspiration before and after the neck operations. However, it was 2 years after the last previous operation of thyroplasty that she progressively developed oropharyngeal dysphagia with frequent coughing and aspiration. Physical and neurological examinations were unremarkable. Endoscopy and cervical computed tomography (CT) revealed no structural abnormality. A videotaped barium radiographic study (videoesophagraphy) showed a narrowing segment at the level of the cricopharyngeus muscle with partial dilation of the proximal esophageal segment (Fig. ).
*A bottle of liquid barium sulfate was diluted with 60 ml of water
After cricopharyngeal dysfunction was confirmed by barium radiographic study, operation was arranged for the patient in which cricopharyngeal myotomy was done (Fig. ). Immediately after the operation, the dysphagia resolved remarkably. Postoperative videoesophagraphy (Fig. ) showed improved cricopharyngeal opening, and there was no sign of the cricopharyngeal narrowing. The patient has remained asymptomatic for 4 years of follow-up, and no adverse side effect of the myotomy has been complained by the patient.
*A bottle of liquid barium sulfate was diluted with 60 ml of water


The patient was a 32-year-old male ex-smoker. He was diagnosed as MFS at the age of 26 based on an aortic dissection (Stanford type A), aortic regurgitation and marfanoid habitus. A Bentall operation with an artificial valve replacement for aortic dissection and a regurgitation of the aortic valve was performed. Thereafter, he was required to take warfarin for the artificial heart valve and was followed at a local hospital. His adherence to medication was poor, which went unnoticed, resulting in the dose of warfarin gradually being increased to 6.5 mg per day in 2017, when he was admitted to the emergency unit of our hospital suffering from a massive haemoptysis and dyspnoea.
The patient had a history of left pneumothorax at the age of 16; however, the details of this are unknown since his mother died in his childhood and the father has been missing since then. On presentation, the patient was fully conscious with a height of 198 cm, body weight of 77 kg, blood pressure of 105/56 mmHg, pulse rate of 90 bpm, body temperature of 36.9 °C, and a saturation of percutaneous oxygen (SpO2) of 94% in room air. The physical examination revealed coarse crackles on auscultation, especially in the right upper lung areas. He had characteristic features such as a tall height, arachnodactyly, dolichocephaly and retrognathia, which are compatible with MFS.
Laboratory data demonstrated anaemia (blood haemoglobin: 10.0 g/dL), an elevated white blood cell count (12,300 /μL), a prothrombin time of 32.5 s (international normalized ratio PT-INR: 5.21), and an active partial thromboplastin time of 61.2 s. Serum tumour markers, antibodies of connective tissue disease and markers of infection such as Aspergillus and Mycobacterium were all negative. Pathological bacteria were not detected in sputum culture. A chest X-ray showed a right apical pulmonary cyst with an air-fluid level inside and consolidation in the right lower lung field (Fig. b). A chest computed tomography (CT) scan indicated multiple cystic lesions in the upper lobes (Fig. b) and a maximum intensity projection (MIP) detected an abnormal vessel originating from the right subclavian artery, which extended into the cyst (Fig. ).
An emergency bronchoscopy revealed continuous bleeding from the apical segmental bronchus of the right lung (B1) (Fig. ), which appeared to have been stopped by topical application of epinephrine and thrombin. Vitamin K and haemostatic medications were infused, which was followed by a transfusion of four units of blood. After emergent therapy, the patient was transferred to the cardiovascular unit of another hospital. An angiography was performed and the abnormal vessel was radiographically diagnosed as the LTA (Fig. a, b). Moreover, the bleeding was stopped successfully by embolization of the artery, which confirmed that the abnormal vessel responsible for bleeding was the LTA. The patient’s condition improved, and warfarin was restarted with strict control of PT-INR. The patient has since been stable with no signs of re-bleeding.


The patient was a 53-year-old male.
Chief complaint: Left lower quadrant pain
Past history: Right inguinal hernia (surgery at age 2), prostatic hypertrophy
Family history: Nothing in particular
History of present illness: He made an emergency visit to our hospital due to left lower quadrant pain since 3 days ago, and it gradually exacerbated.
Present status at admission: Height 165 cm, weight 60.5 kg, and BMI 22.3.
His vital signs were normal: JCS0, blood pressure 137/81 mmHg; heart rate 92/min; body temperature 36.7 °C; and SpO2 99% (room air). His abdominal region was slightly swelling and soft. He felt pain upon applying pressure on the left hypogastrium. There was no rebound tenderness.
Biochemical examination of his blood at admission: His WBC was slightly high at 10090/μl, but his CRP was not high at 0.23 mg/dl. An arterial blood gas analysis did not reveal acidosis (Table ).
Findings upon abdominal enhanced CT: The bladder left side wall was inverted, where part of the enlarged small intestine was found. While contrast enhancement of the small intestine was observed, ascites were present in the space between the enlarged small intestine and the bladder (Fig. ).


A 53-year-old Caucasian man was admitted to our hospital with back pain in March 2016. He was a heavy smoker with a 30-pack-year smoking history. He had had hypertension for 2 years. Regarding his medical history, there was nothing in particular to note apart from metastatic renal cancer and its complications. Indeed, on physical examination, the patient appeared well.
The patient underwent computed tomography (CT), which showed a mass in the left kidney (75 × 53 × 105 mm), as well as lung and liver metastases, confirmed by magnetic resonance imaging examination.
He underwent hepatic biopsy with a negative result because of exiguity of the sample. The patient received left radical nephrectomy and splenectomy in April 2016, and postoperative histopathology revealed clear cell carcinoma of the kidney. In May 2016, the patient was admitted to our hospital again because of kidney failure and electrolyte alterations.
A CT scan at that time confirmed lung and hepatic metastases, and the patient received supportive care with resolution of metabolic alterations (Fig. a).
In June 2016, he started treatment with pazopanib 800 mg daily for metastatic renal cancer.
After 3 months, he had a repeat CT scan (Fig. b), which revealed evidence of necrotizing metastases with cavitation on the lung similar to abscesses in the absence of fever. He stopped pazopanib and started therapy with antibiotics as suggested by a pneumologist.
One month after starting antibiotics, a CT scan showed resolution of the patient’s abscesses but progressive disease (Fig. c).