A 20-year-old G1P1 Hispanic female in previously good health with a history of pyelonephritis during pregnancy and eczema presented with 3 d of sharp, intermittent right lower quadrant pain that radiates towards the midline with movement. She vomited non-bloody, non-bilious emesis three times the day before presentation after eating and presented to our emergency department after having acute appendicitis ruled out at an outside hospital three days prior. Her pain responds to ibuprofen. She denies fevers, chills, diarrhea, urinary urgency or hesitancy, vaginal discharge or bleeding, or trauma. She engages in monogamous unprotected sex with a male partner and endorses mild dysuria. She denies alcohol, tobacco, or other drug use. She experienced a similar episode of pain a couple months prior to presentation. Her only medications are intramuscular depo provera for birth control and ibuprofen for her abdominal pain. She is afebrile with vitals within normal limits and body mass index of 19. Abdominal examination revealed present bowel sounds, mild tenderness to the right of midline near the right lower quadrant without guarding, McBurney’s point tenderness or Murphy’s sign. The remainder of the exam, including a pelvic exam, was unremarkable. Abdominal and pelvic causes of pain including acute appendicitis, nephrolithiasis, pregnancy, tuboovarian abscess, ovarian torsion, small bowel obstruction, functional intestinal obstruction, urinary tract infection, sexually transmitted infection, muscle strain were considered. Transvaginal and limited abdominal ultrasound performed at the outside hospital demonstrated small bilateral follicular cysts and no evidence of appendicitis or other tuboovarian processes. Urine studies were negative for bacteriuria, leukocyte esterase, nitrite, and both gonococcal and chlamydial RNA. Urine cultures acquired at that time were eventually negative for growth. Urine beta-hCG was negative. Wet mount unremarkable. Abdominal X-ray was unremarkable with nonspecific bowel gas pattern. At this point her signs/symptoms and lab testing were unremarkable for acute pathology other than perhaps abdominal pain secondary to follicular cysts. She was discharged in ambulatory, stable condition with instructions to present to pediatric acute follow-up clinic in 2-3 d and pain control with ibuprofen.
The next day she presented to the pediatric acute follow up clinic with severe and constant right lower quadrant pain that was no longer responsive to ibuprofen, and is exacerbated with any ambulation or lifting her child. She is afebrile with normal vitals. Abdominal exam is remarkable for right lower quadrant tenderness to palpation, guarding, and Rovsing’s sign with no rebound tenderness. The patient also clarifies that her right lower quadrant pain has been intermittent and ongoing for the last four months. Hemoglobin (13.1 mg/dL), white blood count (7100/μL), platelets (260000/μL), fibrinogen (303 mg/dL) were within normal with a blood differential notable for peripheral eosinophilia (18% eosinophils with 1300/μL absolute count). Inflammatory markers C-reactive protein (< 0.02 mg/dL) and erythrocyte sedimentation rate (8 mm/h) were normal. Amylase (82 U/L) and lipase (24 U/L) were within normal limits and pelvic as well as right upper quadrant ultrasounds both unremarkable for acute pathology. CT scan of the abdomen and pelvis is unremarkable, particularly demonstrating an air-filled appendix within normal limits and no fat stranding. She is admitted to the pediatrics ward and a gynecology consult determined that a gynecological problem is unlikely. Pediatric surgery was consulted and a diagnostic laparoscopy found normal liver, intestines, uterus, bilateral ovaries and fallopian tubes. There were anomalous adhesions of the cecum to the anterior abdominal wall and the appendix was mildly injected with some area of thickening and induration suspicious for chronic appendicitis, and thus resected.
Her immediate post-op period was unremarkable and she was discharged with appropriate incisional tenderness on post-op day two. She presented to the emergency department on post-op day five complaining of right lower quadrant pain similar to that prior to surgery. She was readmitted and further imaging and workup including pelvic ultrasound as well as MRI of the brain and spine, and failed to show a cause of her pain. Her pain was managed with non-steroidal anti-inflammatory drugs, intravenous morphine, and Norco while in hospital. Surgical pathology of her appendix was grossly unremarkable, without ulceration or inflammation, yet demonstrated histiocytes in lymphoid aggregates on H and E stain and suspicious for LCH. This was confirmed with S-100, CD1a, and CD207 reactivity (Figure ). Upper and lower endoscopy demonstrated whitish-yellowish polypoid nodules less than 5 mm in size at the luminal surface of the cecum, transverse, and descending colon (Figure ). Microscopic evaluation demonstrated similar morphology to the appendix H and E, and immunohistochemical analysis demonstrated S-100, CD1a, CD207 (langerin) reactivity of these nodules (Figure ) whereas biopsy samples of the esophagus, stomach, duodenum, and rectum failed to show histiocytosis. Bone marrow aspirates, bone scan, and MRI brain were negative for bone marrow, skeletal, and pituitary involvement, respectively.


A 45-year-old woman, renal transplant recipient, presented to us with fever with chills, myalgia, severe headache, and persistent vomiting of 6 days duration. She had undergone deceased donor renal transplant 4 years ago, and did not have any rejection or major infections. She was on triple immunosuppression (tacrolimus – 3 mg/day, mycophenolate mofetil – 1500 mg/day, and prednisolone – 7.5 mg/day) and was maintaining normal graft function. There was no pallor, icterus, lymphadenopathy, rash, or edema. Physical examination revealed pulse rate of 102/min, respiratory rate of 16/min, temperature of 103°F, blood pressure of 120/80 mm Hg, and a circumscribed eschar on the back just below left scapula []. Examination of cardiovascular, respiratory system, and abdomen was normal. Neurological examination revealed neck stiffness, mild irritability with normal mentation, and optic fundus. She was started on oral doxycycline of 100 mg twice daily after clinical diagnosis of scrub typhus meningitis was made. Laboratory investigations revealed urine analysis: Trace proteinuria; total blood count: 6500 cells/mm3 with a polymorphonuclear predominance; hemoglobin: 11 g/dl, platelet count 100,000 cells/mm3; normal peripheral smear; blood urea 60 mg/dl; serum creatinine 1.6 mg/dl; bilirubin 0.8 mg/dl; alanine aminotransferase (ALT) 53 IU/L; aspartate aminotransferase 48 IU/L; alkaline phosphatase 86 IU/L; total protein 6.8 g/dl; and albumin 3.8 g/dl. IgM antibodies against O. tsutsugamushi by enzyme-linked immunosorbent assay (ELISA) were positive. Blood and urine cultures were negative. Quantitative buffy coat for malaria, microscopic agglutination tests, and IgM and IgG antibodies for leptospirosis and dengue were negative. Blood polymerase chain reaction (PCR) for cytomegalovirus was negative. Chest X-ray and electrocardiogram were normal. Abdominal ultrasound showed no organomegaly and normal allograft kidney. Computerized tomography of the brain was normal. Cerebrospinal fluid (CSF) analysis showed mild pleocytosis (6–7 cells) mainly lymphocytes, high protein (203 mg/dl), normal sugar (77 mg/dl), normal adenosine deaminase (6 U/L), and negative for tuberculosis and herpes simplex virus by PCR. The patient had persistent fever and headache even after 5 days of oral doxycycline therapy. She was started on IV azithromycin 500 mg/day. She became afebrile within 24 h, her general condition improved dramatically and discharged with normal creatinine. She received totally 1 week of IV azithromycin followed by oral dose for 1 week.


An approximately 13-week-old, male, entire, domestic shorthair kitten weighing 1.59 kg was surrendered to a Brisbane clinic. Physical examination was unremark-able. After a 1 week isolation period, the kitten was castrated, microchipped, treated for parasites, vaccinated and adopted by a member of the public.
Six days later the kitten presented to the clinic with a 1 day history of vomiting, anorexia and lethargy.
The kitten had lost 170 g since adoption and was subdued. Anisocoria was noted with a mydriatic right pupil. No other significant abnormalities were identified. The kitten was started on once-daily subcutaneous amoxicillin/clavulanate (Clavulox; Pfizer) injections at 8.75 mg/kg.
Results of haematological and biochemical testing, pre- and postprandial serum bile acid concentrations and in-house urinalysis are presented in and . Absolute lymphopenia and moderate monocytosis were identified. Testing for feline leukaemia virus antigen and feline immunodeficiency virus antibodies was negative (SNAP FIV/FeLV Combo Test; IDEXX).
On day 3 of hospitalisation the kitten developed vertical nystagmus and ataxia, and was euthanased.
Necropsy demonstrated a 1.5 × 1.5 cm dark brown-to-black soft area that predominantly involved the left parietal lobe and extended across the longitudinal fissure to include a 0.5 × 0.5 cm region of the right parietal lobe of the cerebrum (). On cut surface the lesion extended 1 cm into subcortical white matter. The left cerebral hemisphere was swollen with flattening and widening of gyri in comparison. The meninges were diffusely slightly reddened. No other gross lesions were identified on examination of the nasal cavity, sinuses, thoracic or abdominal cavities. Histopathology was performed on the brain.
Histological examination ( and ) showed fulminating, focal, pyogranulomatous meningoencephalitis with intralesional pigmented fungus, consistent with phaeohyphomycosis. Leptomeninges and Virchow–Robin spaces were expanded by severe infiltrate of macrophages, lesser neutrophils, lymphocytes, multinucleate giant cells and occasional plasma cells. Leukocytes were often centred on golden-brown pigmented fungi, present extracellularly and within the cytoplasm of macrophages and multinucleate giant cells. Fungal hyphae were 2–5 µm in diameter, poorly septate, sparsely and non-dichotomously branched, and had thin, slightly bulbous walls. Conidiophores were 2–10 µm, oval, present in chains (pseudohyphae), and sometimes showed a thin, translucent capsule. In severely affected areas, leukocytes extended into the adjacent white or grey matter parenchyma. Such regions occasionally showed gliosis, gemistocytic astrocytes, rarefaction and malacia. Rare meningeal veins had fibrin thrombi, rarely containing fungal elements.
Brain tissue was plated on Sabouraud agar and incubated at 28°C for 28 days, until growth of an olivaceous-to-greyish-green woolly fungus was detected. The microscopic morphology showed dark septate hyphae with conidiophores producing long chains of oval conidia. The fungus was given a presumptive identification of Cladosporium species and referred for molecular identification.
DNA sequence analysis of the internal transcribed spacer (ITS) 1, 5.8S and ITS2 regions, and the D1/D2 region of the 28S (large subunit) ribosomal DNA gene cluster, using published primers and standard sequencing methodologies identified the fungus as C bantiana. The isolate’s sequence was compared with those in the ISHAM ITS database and the CBS database using the BLASTn search tool and showed >99.8% identity to MITS1144 and CBS118738, respectively.


A 16-year-old neutered female domestic shorthair cat was referred for neurological examination after approximately 2 years of reduced exercise and 2 weeks of reluctance to jump, stiffness in tail movements and lower back discomfort. The cat was up to date with vaccinations and had always been an indoor cat. There was no history of trauma or previous illness, apart from chronic renal disorder, which had been controlled with benazepril.
General physical examination was unremarkable. On neurological examination mild pelvic limb ataxia, reduced perineal reflex and lumbosacral pain on palpation of the spine were present. The presumptive anatomical localisation was either to the caudal lumbar intumescence (L6–S3) or to the cauda equina. Differential diagnoses included a space-occupying lesion, such as neoplasia or a protruding disc, as well as vascular and infectious/inflammatory diseases.
A complete cell blood count revealed reduced red blood cell count 3.5 × 1012/l (reference interval [RI] 7.00–11.60), haemoglobin 5.8 g/dl (RI 9.0–14.0), haematocrit 0.170 l/l (RI 0.29–0.46) and lymphocytes 0.86 × 109/l (RI 1.5–8.0). Serum biochemical abnormalities included increased urea 17.2 mmol/l (RI 6.0–10.0), creatinine 212 μmol/l (RI 40–150), glucose 12.5 mmol/l (RI 4.3–6.6), cholesterol 5.0 mmol/l (RI 1.9–3.9) and creatine kinase 385 IU/L (RI 0–152). On analysis, urine was poorly concentrated (specific gravity 1.016) and proteine/creatinine ratio was increased 0.3 (RI <0.2). Further investigations for infectious diseases were not performed; however, given the history, the laboratory changes were more likely to be related to the chronic renal disorder.
The cat underwent magnetic resonance imaging (MRI) examination (Aperto, 0.4T; Hitachi Medical System) of the lumbosacral spine. A rounded structure at the level of the L7–S1 right epidural space, apparently arising from the right articular facet joint, was identified. The lesion was well defined, hyperintense to spinal cord parenchyma on T2-weighted (T2W) images and hypointense on T1-weighted (T1W) images, did not exhibit contrast enhancement and was therefore compatible with a cyst-like structure (). Significant displacement of the cauda equina dorsally and to the left was associated with the lesion. Furthermore, at the same vertebral level, the intervertebral disc (IVD) showed decreased signal intensity, and signs of degeneration with osteophytic reaction on the ventral edges of the vertebral bodies were present adjacent to the disc space.
Surgical excision of the cyst was performed. After premedication with methadone (Comfortan 0.2 mg/kg IV; Dechra) and dexmedetomidine (Dexdomitor 1 µg/kg IV; Orion Pharma), anaesthesia was induced with alfaxalone (Alfaxan 1 mg/kg IV; Jurox) and maintained with isoflurane (1.5–2.0%) in oxygen and mechanical ventilation, maintaining an end-tidal CO2 of 33–35 mmHg. Compound sodium lactate solution (Hartmann’s Lactated Ringers 5 ml/kg/h; Braun Vet Care) was administered throughout surgery. Intraoperative analgesia was provided by administration of alfentanil (Rapifen 0.00075–0.001 mg/kg/min; Janssen). Cefuroxime (Zinacef 20 mg/kg IV; GlaxoSmithKline) was administered once immediately before surgery. After L7–S1 dorsal laminectomy, a clear fluid-containing cyst-like structure of approximately 3 mm in diameter located between the right L7 nerve root and cauda equina was identified, corresponding with the reported MRI findings. The wall of the lesion was thin and fragile, and clear and colourless fluid leaked out prior to excision. The cyst protruded ventrally from the vertebral canal and appeared to be in contact with the ventral longitudinal ligament; no communication with the articular facet joint cavity was found. The cyst-like lesion was then entirely excised and samples of the wall were submitted for histological analysis. Preservative-free morphine (Morphine preservative free 0.1 mg/kg; Martindale Pharma) was splashed over the dura mater immediately prior to wound closure. Postoperative medications included buprenorphine (Vetergesic 0.02 mg/kg q6h IV; Ceva) and ketamine (Narketan constant rate infusion 0.2–0.3 mg/kg/h; Vétoquinol) for the first 12 h after surgery and gabapentin (10 mg/kg q8-12h; Summit Veterinary) for 2 months.
Histopathology revealed thick disorganised sheaths of fibrocollagenous tissue, collagen necrosis and infiltration of lymphohistiocytes and siderophages alongside more vascularised parts (). The luminal part of the wall was lined with flattened mesenchymal cells (). The lesion was therefore consistent with a cyst, with an intraspinal ganglion cyst being considered the most likely diagnosis, based on previous classification. The cat recovered uneventfully, showing rapid and progressive signs of improvement, and was discharged 3 days after surgery. At 1 month follow-up the cat was showing occasional episodes of discomfort and mild stiffness on pelvic limbs; no neurological deficits were present on neurological assessment performed 2 and 3 months later.
One month after surgery, the cat presented with suspected visual impairment and ophthalmological examination revealed the presence of bilateral retinal detachment and partial reattachment. Toxoplasma serology was negative and systemic hypertension was confirmed by measuring the systolic blood pressure using Doppler sphygmomanometry (mean reading 210 mmHg). Treatment with amlodipine (Amlodipine 0.625 mg/cat q24h; Summit Veterinary) was initiated, with improvement of the ocular condition and normalisation of the blood pressure 3 weeks later (mean reading 135 mmHg). Seven months after surgery, the cat was euthanased for acute deterioration of the renal condition. At that time, no neurological deficits were reported by the owner.


An indoor 9-year-old castrated male domestic cat was referred to the Veterinary Teaching Hospital (University of Montreal) with a 4 month history of increased upper airway noise. A 2 week course of amoxicillin/clavulanic acid resulted in no clinical improvement and the cat was referred for computed tomography (CT) imaging and rhinoscopy.
On physical examination, there was no ocular or nasal discharge, or nasal deformity. The noise the owners reported was heard upon inspiration and originated from the nasal cavity or nasopharynx. The cat presented a breathing pattern compatible with inspiratory dyspnea with marked stertor. Ocular retropulsion and fundic examination were normal. Flow through nasal air columns was normal bilaterally and no polyp was seen on otoscopic or oral examination. A left parasternal grade II/VI holosystolic murmur was ausculted. Complete blood count and serum chemistry revealed a moderate eosinophilia (1600 cells/μl [range 100–790 cells/μl]) and hyperglycemia (16.08 mmol/l [range 3.95–8.84 mmol/l]). Fructosamine was measured and was normal (256 µmol/l [range 240–420 µmol/l]).
After induction of general anesthesia, a CT scan (16-slice helical; GE Lightspeed) of the nasal cavities, neck and chest revealed a well-defined homogeneous soft tissue mass measuring 2.1 cm in length by 1 cm in width that occupied the entire nasopharynx (), caudal to the nasal choanae. The caudal aspect of the mass extended to the most caudal aspect of the soft palate. Following administration of intravenous contrast medium (12 ml) (Isovue 300; ER Squibb), there was a mild enhancement of the contour, as well as heterogeneous uptake within the mass. A small amount of fluid was visible within the two compartments of the right tympanic bulla. A moderate amount of fluid was present inside the nasal cavities at their rostral and middle thirds. The presence of fluid was symmetrical between the two nasal cavities and was associated with a decrease of the normal contour, enhancement and number of the nasal turbinates. A small amount of fluid was also visible in the rostroventral aspect of left frontal sinus. Regional lymph nodes were normal. Diagnosis following CT scan was a nasopharyngeal mass most compatible with a polyp and associated with mild right otitis media and bilateral chronic inflammatory rhinitis. CT scan of the thorax was normal.
Following identification of a nasopharyngeal mass on CT scan, nasopharyngoscopy was performed using a 3.5 mm flexible endoscope (Olympus BF-160 5.3 mm). A well-defined pedunculated smooth, shiny, yellow-brown nasopharyngeal mass was identified caudal to the nasal choanae. An elliptical retrieval basket forcep was introduced through the working channel of the scope in order to grasp the mass. The forceps were inserted through the channel prior to retroflexion of the scope into the nasopharynx. Upon inspection, the mass was determined to be attached by a stalk to the left aspect of the nasopharynx, dorsal to the soft palate, and its removal was possible by grasping it at the level of the peduncle. The macroscopic appearance of the mass was most compatible with a polyp (). Severely inflamed nasal choanae were apparent after withdrawal of the mass. Bilateral rostral nasal biopsies were performed at the end of the procedure and immediately placed in neutral buffered formalin for histopathological analysis along with the mass.
Histopathology revealed that the mass was compatible with polypoid granulomatous pharyngitis. The polypoid mass was covered by a stratified squamous epithelium and was composed of a collagenous stroma severely infiltrated by macrophages associated with some clusters of neutrophils. Numerous round organisms with thick capsules (‘bubble soap’ appearance) measuring 10–30 μm in diameter were present in the cytoplasm of macrophages or free in the stroma and were compatible with fungi. Periodic acid–Schiff and Grocott’s methenamine silver stains revealed the presence of bodies with a narrow budding, suggestive of Cryptococcus (). Biopsies of the nasal turbinates revealed the presence of few neutrophils and eosinophils in the lumen of blood vessels and in the lamina propria without fungal elements in the tissues examined. A serum latex cryptococcal antigen agglutination test (LCAT; New York State Veterinary Laboratory, Cornell University, Ithaca, New York), was performed and a strong positive antigenic titre of 256 was found.
Following excision of the polypoid mass, audible stertor resolved and the respiratory pattern of the patient returned to normal. Treatment with itracona-zole (Sporanox 100 mg capsules; Janssen-Cilag) was initiated at a dose of 50 mg q24h for 4 months but was discontinued after only 1 month due to client error. Four months after the procedure, no recurrence of clinical signs was observed and a 32× decrease in the LCAT titre (titre of 8) was observed, indicating a positive response despite only 1 month of therapy. Itraconazole was restarted at the same dose for 4 months following the second LCAT titer. One year after diagnosis, the LCAT titer was negative and the cat remained free of clinical signs.


A 10-year-old male neutered British Shorthair cat was presented for chronic lethargy and anorexia, as well as acute polyuria/polydipsia and faecal incontinence. The cat’s general condition had deteriorated over the previous 6 months, with notable weight loss. Progressive alopecia had also been noticed by the owners. The cat was housed individually and had free outdoor access. The cat was negative for feline leukaemia virus status and feline immunodeficiency virus. Previous unsuccessful therapy consisted of antimicrobials and corticosteroids of unknown type and dose.
At clinical examination the cat was thin (body condition score 2/9) and had a bilateral purulent ocular discharge. Alopecia extended symmetrically from the ventral abdomen to the medial thighs. The skin in the alopecic areas was erythematous, smooth and glistening (). Crusts and brown exudate were found between the toes. Spontaneous pruritus was present. Microscopic evaluation of impression smears taken from the abdomen and digits revealed a high amount of cocci and Malassezia species yeast. Ectoparasites could not be detected in multiple superficial and deep skin scrapings. Trichograms and Wood’s lamp examination were negative for dermatophytosis.
The cat was hospitalised and started on intravenous fluids. To treat pyoderma and Malassezia species dermatitis amoxicillin/clavulanic acid (20 mg/kg q12h IV, AmoxClav; Hexal AG) and itraconazol (5 mg/kg q24h PO, itrafungol; Elanco Animal Health) were administered.
Haematology and serum biochemistry showed only mild changes, which were considered clinically insignificant. Urinalysis showed a mildly increased urine proteinto creatinine ratio (0.32) and an inactive sediment. During the abdominal ultrasound approximately 1.5 cm of the colonic wall appeared hypoechogenic and thickened with loss of normal layering. Additionally, there were signs of mild focal peritonitis. Multiple target lesions in the liver and enlarged and hypoechogenic abdominal lymph nodes were suggestive of tumour metastases. Fine-needle aspirates were taken from lymph nodes, liver and the altered colonic wall. All samples contained atypical cells of the same type with signs of malignancy, including anisocytosis, anisokaryosis, pleomorphism, light basophilic cytoplasm and few atypical mitotic figures (). All samples were of high cellularity and contained spindle-shaped to polygonal cells located in small clusters or as singletons. The cells possessed eccentrically located round-to-oval nuclei with reticular chromatin pattern, 1–2 often prominent nucleoli and moderate amounts of a lightly basophilic cytoplasm with several fine intracytoplasmatic vacuoles, as well as distinct cellular borders. The cytological picture was consistent with a malignant undifferentiated tumour; however, determination of the histogenesis was not possible. Owing to the poor prognosis the owners elected to have the cat euthanased. At necropsy the abdominal skin was diffusely alopecic and on the paws hyperaemic erosions were seen. A 2 cm × 1 cm × 0.3 cm ulcerated neoplasia of the dorsal colonic wall was found (). In this localisation there was a chronic peritonitis with fibrous adhesions involving the small intestine. On the serosal surface numerous dilated lymphatics and marked desmoplasia were noted. The omentum contained multiple disseminated firm white small nodules (). Similar nodules of 0.5–1.0 cm in diameter were found within the mesenteric lymph node, liver and diaphragm ().
Histopathology of the skin showed moderate hyperplasia and acanthosis, mild ortho- and parakeratotic hyperkeratosis with some yeasts between keratin layers and mild superficial pyoderma. Hair follicles were atrophic or predominantly in telogen phase of the hair cycle (, ). Sebaceous glands were unaltered. A few dilated infundibula filled with neutrophils and detritus (suppurative folliculitis) were detectable at the muzzles.
The intestinal neoplasia was identified as tubular-to-anaplastic carcinoma with desmoplasia and lymphatic invasion (). Metastases were found within mesenteric lymph node, peritoneum and liver. Immunohistochemistry using pan-cytokeratin AE1/2 (PAP method) determined the epithelial origin of the neoplastic cells.


A 14-year-old, female, spayed, domestic shorthair cat weighing 5.68 kg presented on referral to our hospital for evaluation of acute collapse. The cat had a history of diabetes mellitus (DM), which was first treated with GI in 2013. The cat was treated for diabetes, which became transient after 3 months of treatment. One week prior to presentation at our institution, the DM had recurred and re-initiation of GI was recommended. The owners had purchased a new bottle of GI from a local pharmacy. The bottle had not been opened prior to use and it was stored in a refrigerator. The evening of presentation the cat had ingested all of its meal (which had not been changed in over a year), according to the owner, and received 1 unit of GI subcutaneously for the first time since 2013. Approximately 2–3 mins after administration, the cat collapsed and started panting. The cat also demonstrated projectile vomiting and diarrhea. On presentation to the primary veterinarian, the cat was noted to be laterally recumbent and was exhibiting horizontal nystagmus. A blood glucose (BG) was not obtained, but the cat was given dextrose submucosally as there were concerns about a hypoglycemic crisis. The cat was then referred to our facility.
On presentation to our institution, the cat was noted to be obtunded, was tachycardic (heart rate of 220 beats per minute) and tachypneic (respiration of 50 breaths per minute). Rectal temperature was normal (101.2ºF). The respiratory effort was increased and infrequent stridor noted. The face and muzzle were swollen and edematous. The cat again produced a large amount of vomiting and diarrhea; at this time hematochezia was also noted. Though mentation was dull, the nystagmus had resolved and no cranial nerve deficits were noted.
BG was measured on presentation with a handheld glucometer (AlphaTrak; Abbott Animal Health) and was noted to be 517 mg/dl (reference interval [RI] 75–116 mg/dl); this was approximately 2 h after the GI had been administered. The cat had no measurable systolic blood pressure on Doppler measurement (Parks Doppler Machine) and a 10 ml/kg bolus of crystalloids was administered. Systolic blood pressure measured after the crystalloid bolus was noted to be 120 mmHg. Owing to the concerns about an HR to the insulin, diphenhydramine (2 mg/kg) was administered intramuscularly. Within an hour, the muzzle swelling and erythema had improved after antihistamine administration. The cat continued to experience recurrent systemic hypotension in the subsequent hours and after receiving a 3 ml/kg synthetic colloid bolus, was started on a continuous crystalloid (75 ml/kg/day) and colloid infusion (8 ml/kg/day) and the systemic hypotension resolved. Blood work performed on admission revealed hyperglycemia (666 mg/dl; RI 75–116 mg/dl), hypokalemia (3.36 mEq/l; RI 3.62–4.60 mmol/l) and hyperlactatemia (5.4 mmol/l; RI 0.7–2.8 mmol/l). The serum was evaluated for ketones at presentation and none were noted. A complete blood count was also performed and was unremarkable.
Treatment for her gastrointestinal signs consisted of maropitant (1 mg/kg SC q24h), dolestron (0.6 mg/kg IV q24h) and ampicillin (20 mg/kg IV q8h).The cat continued to experience a large volume of diarrhea and hematochezia over the first 24 h after admission, but the vomiting resolved.
An abdominal ultrasound was performed 1 day after presentation and revealed an enlarged and hyperechoic liver with few small hypoechoic nodules throughout the parenchyma, assessed as consistent with a diabetic hepatopathy. The gallbladder was within normal limits. Both kidneys had a mild decrease in corticomedullary definition, and the left and right adrenal glands were noted to have normal shape and size. No other abnormalities were noted. Blood work was also repeated at this time and showed normalization of the lactate and potassium levels. During hospitalization, the BG continued to be monitored and ranged from 107–355 mg/dl; the cat’s appetite remained poor. No insulin was administered while hospitalized, owing to the poor appetite, persistent diarrhea and the concerns about an HR to GI. Serum ketones were monitored daily and continued to be negative. The cat was discharged 3 days after presentation on metronidazole (10 mg/kg PO q12h) and famotidine (1 mg/kg PO q24h). The owners continued to monitor the cat’s BG at home with a handheld glucometer and it ranged between 88 and 380 mg/dl. The owners tended to measure the BG if they felt the cat appeared lethargic; they did not have a set protocol of when and how frequently to measure its BG. At this time the cat was still not consuming its full caloric needs and we recommended that the owners continue to monitor the cat’s BG at home to determine when it would require insulin again. The owners infrequently measured the cat’s BG 1–2 times weekly and it was never greater than 200 mg/dl.
Six months later, the cat re-presented for recurrence of polyuria/polydipsia and being lethargic at home. The owners were continuing to measure the cat’s BG at this time and were intermittently obtaining values greater than 250 mg/dl. Fructosamine was found to be elevated (400 µmol/l; RI 142–450 µmol/l), and the cat was also exhibiting intermittent glucosuria. The cat was sedated with ketamine (0.5 mg/kg IV) and valium (0.3 mg/kg IV), and the fur over the lateral left thorax was clipped. One unit of GI and porcine zinc insulin (PZI) were each diluted into 20 units/0.2 ml of 0.9% NaCl and from each of these mixtures, 0.05 ml/5 units was injected intradermally. As a negative control, 0.05 ml/5 units of 0.9% NaCl was also injected. All intradermal injection sites were approximately 2 cm apart. Within 30 mins of the procedure, a palpable swelling was noted over the GI site. The cat also developed erythema and swelling of its muzzle () and vomited; diphenhydramine (2 mg/kg IM) was administered and the clinical signs resolved. Two hours after the procedure, the cat’s BG was 410 mg/dl (RI 90–180 mg/dl) and it was elected to administer 0.5 units PZI subcutaneously, owing to the hyperglycemia. The cat tolerated the injection of PZI well and no adverse effects were noted. The cat was kept overnight for monitoring and was noted to be painful and intermittently pruritic over the injection site. The cat was discharged the following day and continues to be on subcutaneous PZI every 12 h and has not exhibited any recurrent signs of systemic or cutaneous HR.


A 7-month-old, 3.8 kg intact male Manx cat presented to the Kansas State University Veterinary Health Center for acute onset of vomiting and tenesmus. The cat was previously healthy, being fed Purina Healthy Kitten dry food, but was incompletely vaccinated, with only one feline leukemia virus (FeLV; Purevax Feline Leukemia [Merial]) and FVRCP vaccine (HESKA Feline UltraNasal FVRCP Vaccine; HESKA) at 14 weeks of age. The cat lived strictly indoors, along with one dog.
On initial evaluation, the patient was lethargic, tachycardic (240 beats per minute [bpm]) and tachypneic (80 respirations per minute [rpm]), with apparently normal heart and lung sounds. The cat was febrile (40.8°C), and frequent tenesmus was appreciated throughout the examination. Abdominal palpation revealed a small, soft urinary bladder, and rectal examination was unremarkable.
A complete blood count (CBC), serum biochemistry profile and urinalysis (collected by cystocentesis) were performed on day 1 (see and ). CBC and serum biochemistry abnormalities included an inflammatory leukogram, with a normal total white blood cell (WBC) count, elevated band neutrophils, lymphopenia, hyperglycemia and mildly elevated alkaline phosphatase. Urinalysis revealed a urine specific gravity >1.050, glycosuria (3+) and proteinuria (3+). Owing to the presence of glycosuria, urine was submitted for bacterial culture, which revealed no growth on day 3 of hospitalization. Abdominal radiographs were obtained and interpreted as unremarkable. The patient was hospitalized and started on intravenous (IV) Lactated Ringer’s Solution (LRS) at 25 ml/h for 12 h, which was then reduced to 15 ml/h. In addition, buprenorphine (0.01 mg/kg IV q8h) was administered for perceived pain associated with tenesmus.
The following day, the patient remained lethargic and febrile (40.5°C). Systolic blood pressure was recorded from the right forelimb and measured to be 138 mmHg. An abdominal ultrasound was performed, which revealed a hypoechoic liver and spleen, multifocal lymphadenopathy, and scant peritoneal effusion. The amount of abdominal effusion was insufficient for collection, and cytological evaluation of an ultrasound-guided fine-needle aspirate of the liver revealed mild vacuolar hepatopathy. A CBC revealed an acute inflammatory leukogram with marked leukopenia, characterized by neutropenia, lymphopenia, elevated bands and mild toxic changes in the neutrophils (see ). Significant abnormalities on a serum biochemistry profile performed on day 2 included hypoproteinemia, hypoalbuminemia and elevated creatine kinase (). Treatment with IV LRS (15 ml/h) and buprenorphine was continued. Owing to continued vomiting, maropitant (1 mg/kg SC q24h) was initiated along with ampicillin/sulbactam (22 mg/kg IV q8h) and azithromycin (10 mg/kg PO q24h).
On day 3 of hospitalization the cat remained febrile (40.5°C) and mucous membranes appeared mildly icteric. Systolic blood pressure remained within a normal range of 120–130 mmHg, vomiting and tenesmus had stopped, and the cat was more active and began to eat. Cellulitis was noted on the caudal ventral abdomen extending around the prepuce, scrotum and rectum, and significant pain was elicited when palpating the lumbar–sacral spine. A repeat CBC revealed an improved total WBC, with neutropenia and elevated bands with moderate toxic changes in neutrophils (see ). Significant abnormalities on serum biochemistry revealed a persistent hypoproteinemia, hypoalbuminemia, increased creatine kinase and hyperbilirubinemia ().
The caudal ventral abdominal and perineal cellulitis showed necrotic changes on day 4, with yellow–gray skin discoloration and swelling, with a distinct demarcation between affected and non-affected tissue (). Margins were traced with permanent marker to monitor for progression. The affected area extended 5 cm cranial to the prepuce with 1 cm margins lateral to the scrotum and prepuce, and extended to the lateral and caudal margins of the tail base. In addition, it was noted that anal tone and sensation were decreased. A CBC on day 4 revealed a resolved neutropenia (see ), and serum biochemistry revealed a lower serum total bilirubin (see ). A contrast cystourethrogram was performed to eliminate the possibility of urethral tear as the cause of cellulitis and necrosis, and the urethra was found to be intact. The patient remained febrile (40.6°C); however, owing to a reliable appetite and continued improvement in attitude, IV ampicillin/sulbactam and IV buprenorphine were discontinued. Amoxicillin/clavulanic acid (16 mg/kg PO q12h) was initiated in addition to the previously prescribed azithromycin, and buprenorphine was administered subcutaneously.
On day 5 the patient was normothermic (37.8°C), and the necrotic skin changes were more prominent, with no change in wound size/margins. Owing to wound contamination from fecal incontinence and ongoing diarrhea, it was decided that the patient would remain in hospital for broad-spectrum antibiotic administration (amoxicillin/clavulanic acid was continued as above, azithromycin was discontinued and enrofloxacin [4.7 mg/kg PO, q24h] was initiated owing to continued fecal contamination of the wound) in addition to daily wound management/monitoring. The necrotic/sloughing skin was left in place to allow coverage of the underlying tissue from fecal contamination (). The necrotic tissue eventually completely sloughed off naturally around day 30 of hospitalization (), revealing a well-contracted and granulated wound, at which time the cat was discharged from the hospital for further supportive care at home. The patient made a complete recovery (), and was eventually castrated 71 days after initial presentation. The testicles were sent for histological examination, and were found to be atrophied.


A 1-year-old, 6.4 kg, strictly indoor, castrated male domestic shorthair cat presented to Veterinary Centers of America Central Animal Hospital in Upland, CA, USA, for an evaluation of acute-onset vomiting, tenesmus and lethargy. The cat’s diet consisted of Nutro Natural Choice adult dry cat food, with the canned variety occasionally added in. Examination revealed the cat had a slightly elevated temperature (39.3°C), with tacky mucous membranes and tachycardia (230 bpm). Upon palpating the abdomen, tenesmus was induced with only a clear mucoid discharge produced. The bladder palpated soft and small, and a rectal examination revealed an empty colon with clear mucous on the glove.
Abdominal and thoracic radiographs were unremarkable. An initial in-house abbreviated serum biochemistry profile revealed mild hyperglycemia (156 mg/dl [8.7 mmol/l]; reference interval 75–145 mg/dl [4.2–8.0 mmol/l]), with normal blood urea nitrogen, creatinine, alkaline phosphatase and alanine transaminase (ALT). In-house packed cell volume was 45% and total solids was 7.5 mg/dl. FeLV/FIV SNAP (IDEXX) and Parvovirus SNAP (IDEXX) tests were both negative. The cat was hospitalized and started on LRS (30 ml/h IV), maropitant (0.8 mg/kg SC q24h) and metronidazole (19.5 mg/kg PO q12h). Throughout the night, the patient’s temperature increased (40.1°C), and, owing to perceived discomfort, buprenorphine (0.01 mg/kg IV as needed) was given.
The following day, a CBC (see ) and serum biochemistry (see ) were sent to an outside reference laboratory (Antech Diagnostics, Irvine, CA, USA) (see and ). Similar to case 1, the CBC revealed moderate leukopenia characterized by neutropenia and lymphopenia. Significant abnormalities on serum biochemistry included hyperbilirubinemia and mildly elevated creatine kinase. Urinalysis revealed a urine specific gravity of 1.030, bilirubinuria (3+), hematuria (11–20 red blood cells [RBC] per high-power field [HPF]; reference interval 0–3 RBC/HPF), pyuria (4–10 WBC/HPF; reference interval 0–3 WBC/HPF) and no bacteria. In addition to the previously prescribed metronidazole and buprenorphine, the patient was started on amoxicillin/clavulanic acid (14.6 mg/kg PO q12h), enrofloxacin (5.3 mg/kg PO q24h) and robenacoxib (1.87 mg/kg PO q24h) was administered for 3 days.
On day 3 of hospitalization, the patient was normothermic (38.9°C), more alert and had a better appetite. However, pain could be elicited on palpation of the caudal lumbar area. The following day the patient’s mucous membranes showed evidence of mild icterus, and the rectal temperature was again elevated (40.6°C). An abdominal ultrasound was performed on day 5 and revealed a slightly enlarged and hypoechoic liver, and intestinal hypermotility. Bloodwork was again sent to an outside reference laboratory (ANTECH Diagnostics) on day 5. The CBC revealed a mild leukocytosis with neutrophilia (see ). Significant abnormalities on serum biochemistry included worsening hyperbilirubinemia, mild hypoalbuminemia, mildly elevated ALT, mildly elevated aspartate transaminase and a more significant elevation in creatine kinase (see ).
Beginning on the evening of day 6 and progressing to day 7 of hospitalization, the skin from the medial aspects of both caudal thighs, extending towards the inguinal area and the ventral tail base, appeared yellow and necrotic with a distinct line of demarcation from the adjacent skin. As in case 1, conservative wound management was elected, with the cat remaining in hospital (). On day 10 the oral antibiotics were discontinued and cefovicin (7.5 mg/kg) was administered subcutaneously every 2 weeks until sufficient granulation tissue was present, and the wounds were cleansed as needed with dilute Betadine (Purdue Products L.P., Stamford, CT). The necrotic skin sloughed on day 43 of hospitalization, revealing a bed of granulation tissue. The patient was discharged on day 56 with recommendations to clean the wound as needed, and made a complete recovery.


A previously healthy 52-year-old female was diagnosed with a partially obstructing rectal cancer 8 cm from the anal verge. The patient underwent neoadjuvant chemoradiation followed by a robotic ultra-low anterior resection with a stapled anastomosis and proximal diversion. At 2-month post-operatively, a rectovaginal fistula was noted. The patient underwent a perineal repair of the fistula with gracilis muscle interposition flap. The loop ileostomy was reversed after endoscopic and radiological evidence of fistula healing. The fistula recurred 1 month later necessitating another ileostomy. Four weeks later, the fistula was repaired trans-abdominally as described below.
A 5 cm Pfannenstiel incision was made and extended down to the anterior rectus sheath which was divided transversely. Flaps were created superiorly and inferiorly separating the rectus muscle from the anterior fascia followed by division of the peritoneum in the midline to enter the abdomen. Three 5-mm trocars were placed at the umbilicus, the right and left lower quadrants. After laparoscopic resection of the previous anastomosis and take down/repair of the fistula, a redo ultra-low anterior resection was performed with a transanal inter-sphincteric resection, coloanal pull-through and a hand-sewn anastomosis.
The left rectus muscle was then harvested in a laparoscopic-assisted fashion through the Pfannenstiel incision without longitudinal division of either anterior or posterior rectus sheaths []. The rectus muscle was divided superiorly at the level of the costal margin and inferiorly at the pubis while preserving the deep inferior epigastric vessels. The flap was then rotated and transposed between the rectum and the vagina and held by interrupted sutures. The transverse anterior rectus sheath incision was closed primarily. The total operative time was 4 h while the rectus harvest time was less than 45 min. The patient's post-operative course was uneventful with minimal pain, and the patient was discharged home after 6 days. The patient continued to do well at 4-month follow-up without any evidence of fistula recurrence or hernia formation.


A 13-year-old boy who underwent laparoscopic surgery, probably appendectomy 3 months ago for acute appendicitis was referred for episodic pain in lower abdomen over last 1 month that had been treated with trial of antibiotics and analgesics. There were waxing and waning of these symptoms over the mentioned duration. This episode was sudden in onset, severe in intensity associated with two episodes of non-bilious vomiting and high-grade fever which brought this patient to us. On examination, signs of localised peritonitis were evident in the right upper quadrant. Inflammatory markers were elevated. A clinical diagnosis of residual appendicitis was made, and empirical broad spectrum antibiotics were started.
An ultrasound abdomen was performed, which revealed the presence of inflamed noncompressible, non-peristaltic, tubular structure about 2.5 cm in length and 1.2 cm in diameter located in paracaecal region with a collection of viscous fluid of around 50cc. Findings were confirmed by doing a contrast enhanced computed tomography (CECT) []. After optimising the patient clinically, he was taken to the operating room. Laparoscopy revealed intraperitoneal adhesions in right iliac fossa with purulent collection about 50cc. Following careful adhesiolysis, the inflamed residual tip of appendix of size 4 cm laying in paracaecal position was confirmed. We observed the presence of intact blood supply [] to this portion from adjoining colonic wall, which probably has maintained the viability and thus persistent inflammation. The excised specimen was confirmed histologically as residual portion of the appendix. Post-operative recovery was uneventful; the patient was discharged on the 3rd post-operative day.


A 42-year-old multiparous woman presented with episodes of vague abdominal discomfort predominantly on the left side. Per abdominal examination revealed an ill-defined lump in the abdomen arising out of pelvis stretching beyond the umbilicus more on the left side. On pelvic examination, a pelvic mass that was of around 14 weeks gestational size, immobile, slightly tender, more on the left side was palpable. Her last childbirth was 19 years ago and she had undergone a total hysterectomy for a 20 cm fibroid arising from isthmus of the uterus 6 years back. The hematological and biochemical investigations including CA125 levels were within normal limits. Chest X-ray was normal.
Ultrasonography revealed mixed echogenicity mass in pelvis extending bilaterally, predominantly on left side, bilateral adnexae could not be visualized separately from the mass, the uterus was not seen as she had undergone a total hysterectomy. There was no free fluid in the pouch of Douglas []. The upper abdomen was normal. Possibility of complex ovarian mass most likely cystadenoma of ovary was suggested and the patient was taken up for laparotomy. Peroperatively, a single nodule of 5 cm × 5 cm was also identified in the mesentery. The left ovary was enlarged and homogenous around 10 cm × 14 cm, posteriorly going into the retroperitoneum. Right ovary and tube were healthy looking. Tissue was taken for frozen sections from the retroperitoneum and ovary and possibility of a mesenchymal lesion was suggested. Subsequently, the nodules were excised and bilateral salphingoopherectomy was performed and specimen received in 10% formalin for histopathology. Grossly, the retroperitoneum, left ovary, and mesenteric nodules were gray-white, homogenous in appearance, the cut section showed a whorled gray-white appearance []. The right ovary and bilateral tubes were unremarkable. Several tissue blocks were processed and routinely stained for light microscopy examination.
Histology revealed similar findings from the retroperitoneum, left ovary, and mesenteric masses; interlacing and intersecting fascicles of smooth muscle cells arranged in whorls and having an abundant eosinophilic fibrillar cytoplasm with focal myxoid and cystic change were appreciated []. Areas of hyalinization were also present. There was no atypia or necrosis. Histomorphology was suggestive of ovarian leiomyoma and leiomyomatosis peritonealis disseminata (LPD). On Masson trichome stain, the smooth muscle cells stained red and the hyalinized areas were blue []. The smooth muscle cells were positive for smooth muscle actin (SMA) on immunohistochemistry []. The smooth muscle cells were also positive for estrogen receptor (ER) and progesterone receptor (PR) which further supported the diagnosis.


A 54-year-old man presented to our hospital with a 4-month history of intermittent shortness of breath. Transthoracic echocardiography revealed a mild pericardial effusion and minimal pericardial thickening. CT image of the thorax showed fluid collections within the pericardial sleeves (up to 14 mm) and the bilateral pleural spaces (). He had no apparent history of occupational exposure to asbestos. Although CT scan of the chest and echocardiography were performed, a definitive diagnosis could not be obtained. Whole body F-18 FDG PET/CT scan was performed (1 h after the administration of 465 MBq F-18 FDG, with the subject fasted for 6 h beforehand). FDG-PET/CT images showed a diffuse intense F-18 FDG uptake (SUVmax, 7.5) in the thickened pericardium (, ). There were fluid collections within the bilateral pleural spaces and massive fluid in the abdominal and pelvic peritoneal spaces and systemic edema. No abnormal focus of FDG uptake was seen elsewhere in the body, suggesting a possible site of primary. Thus, a primary tumor of the pericardium, possibly malignant PPM was suspected. In addition, there were no additional hypermetabolic lesions suggesting lymphatic or distant metastatic disease. Primary pericardial malignancy as the cause of constrictive pericarditis was highly suspected from F-18 FDG PET/CT results. The patient’s condition deteriorated, and surgical pericardiectomy under cardiopulmonary bypass (CPB) was performed to relieve the symptoms and confirm the definitive diagnosis. The debulking surgery was performed to remove the thickened pericardium. Pathological examination revealed epithelial type PPM (). He underwent palliative treatment and died 2 months postoperatively.


A 7-year-old Hispanic female with an uneventful birth history presented to our referral institution for workup and management of balance and gait disturbance and an inability to walk that was first noted at 3 ½ years of age. She was noted to have increasing “clumsiness” in the months leading to her inability to walk. She was also noted to have chronic renal disease and liver disease with hepatomegaly and enlarged kidneys in the first 4 years of life. Her inability to walk was previously attributed to severe pain throughout her body.
At 6 years of age, she developed bilateral lower extremity fractures and an upper extremity fracture while attempting to stand. She was noted to have bowing of her lower extremities and elevated alkaline phosphatase and was subsequently diagnosed with rickets that was believed to be the result of chronic kidney disease. Shortly thereafter, she was referred to a local medical center where she underwent liver biopsy, kidney biopsy, and muscle biopsy. Her liver biopsy revealed cirrhosis with minimal chronic inflammation and her kidney biopsy revealed nonspecific glomerular and tubular changes with some parenchymal fibrosis. Her muscle biopsies revealed severe myopathic changes with myofiber atrophy. She was noted to have mitochondrial DNA quantification on her muscle biopsy which was reduced and less than 29% of controls. She was subsequently diagnosed with mitochondrial depletion syndrome.
When her weakness and strength worsened further, she was referred to our institution’s mitochondrial clinic where she underwent additional workup. Extensive metabolic evaluation revealed an elevated succinylacetone which led to a diagnosis of HT-1. Analysis of the FAH gene revealed a homozygous splice mutation known to be associated with HT-1. She ultimately underwent treatment with dietary modification and 2-(2-nitro-4-trifluoromethylbenzoyl)-1,3-cyclohexanedione (NTBC), but she was immediately referred for cadaveric liver transplantation after further workup revealed imaging findings consistent with hepatocellular carcinoma. Following this initial pre-transplant treatment, her condition improved substantially with significant improvement in her amino acid profile, improvement in her mental status, and reduction in her alpha-fetoprotein from 2320 ng/mL to 1585 ng/mL. Despite this metabolic improvement, she required liver transplantation as a result of her hepatocellular carcinoma. Since this lesion was felt to be adjacent to major blood vessels and liver transplantation was not ideal without improved nutrition and rehabilitation, she underwent transarterial chemoembolization for a 1.5 cm × 1.6 cm lesion in her liver (Figure ) after which time she had further reduction in her alpha-fetoprotein to 511 ng/mL. After continued improvement in nutrition and physical conditioning, she ultimately underwent liver transplantation 4 mo after her diagnosis was made. Explant of her liver revealed a yellow-orange and nodular liver, and histology revealed cirrhosis and multinodular well-differentiated hepatocellular carcinoma with bile duct proliferation felt to be consistent with tyrosinemia (Figure ).
Her immediate postoperative course was unremarkable and, following transplant, she had further improvements in her cognitive function, nutrition, and physical conditioning. Within 6 mo, she was ambulating without assistance and attending regular school without difficulty. Her rickets had also improved and resolved with her bone mineral density Z-score in her hip improving from -4.1 prior to transplant to -1.1 fifteen months following transplant. Her renal function improved with her estimated GFR before and 6 mo after transplant noted to be 75 and 121 mL/min per 1.73 m2 respectively. She had no recurrence of tumor on follow-up imaging obtained 2 years following her transplantation, and her alpha-fetoprotein was normal during this time.


A 80-year-old woman presented to the emergency service with fatigue. She did not have any other known chronic diseases. Physical presentation revealed 2 cm palpable enlarged spleen below the costal margin, bilateral small cervical lymph nodes and no hepatomegaly. Her hemoglobin level was 11.2 (12-18) g/dL, platelet count was 260.000 (130000-400.000)/µL,white blood cell count was 115.000/µL (4.200-11.000), neutrophil count was 7780 (1900-8000)/µL, lymphocyte count was 25.440 (0.9-5.200)/µL and LUC (large unstained cells) count was 81.200 (0-0.5)/µL. Her blood chemistry was unremarkable, except for a lactate dehydrogenase level of 243 (125-220) U/L, and beta-2 microglobulin level of 5.3 mg/L (0.7-1.8). Peripheral blood smear evaluation showed 85% prolymphocytes with moderately condensed nuclear chromatin, prominent nucleoli, and a faintly basophilic cytoplasm (). She had no monoclonal gammopathy in the serum and urine immunoelectrophoresis. PET-CT showed mediastinal lymph nodes with cervical lymph nodes. And the greatest size of mediastinal lymph nodes was 20×12 mm and cervical lymph nodes was 8×13 mm. There was no pathological FDG involvement in the spleen. BM aspiration smear exhibit atypical wide lymphocytes with prominent nucleoli and abundant agranular cytoplasm (). There were no cytoplasmic projections such as hairy projections or blebbings on the cell surface of the atypical lymhocytes ( and ).
Flow cytometry analysis revealed CD34 negative lymphocytes. Lymphocytes had negative CD33-, CD13-, CD117-, CD23-, FMC7-, MPO-, CD15-, CD14-, CD64-. And lymphocytes had positive CD5+, CD19+, CD20+, CD22+, CD11c+, CD25+, CD79a+ and CD79b+. Fluorescence in situ hybridization (FISH) technique analysis reveals no translocation between the 11th and 14th chromosome. BM biopsy revealed interstitially distributed atypical cells with wide nucleus and prominent nucleolus (). Immunohistochemical staining of the atypical cells showed diffuse CD20, PAX5, CD5, BCL2 positivity and partial CD23 strong positivity (). TDT, CD10, CD34, CD117, BCL1, BCL6, SOX11 and Annexin were not detected on the atypical cells. Besides, CD25 was weakly and non-specifically detected by staining. Lower rate of lymphocytes with small size were detected to have CD3 by staining. Lastly, reticulin fiber was not increased (grade 0/3). BM biopsy was reported as primarily compatible with prolymphocytic leukemia for the diagnosis.


A 28-year-old male presented with raised intracranial pressure for two months. Neurological examination of the patient revealed bilateral papilledema. However, there were no focal deficits. MR of the patient revealed T1 hypointense, T2 hyperintense intraventricular lesion filling the bilateral lateral ventricle with avid contrast uptake and central non-enhancing area [Figure –]. There was a non-enhancing area in the right frontal lobe contiguous with the intraventricular lesion as apparent on T2 and FLAIR images []. Patient underwent a parasagittal craniotomy and near total excision of the tumor. The tumor was pinkish gray, very vascular and had areas of necrosis along with thrombosed vessels within. Septum pellucidum was involved and was resected partly along with the tumor. The floor of both lateral ventricles could be appreciated after resection. The right frontal lobe appeared normal intraoperatively. Histopathological evaluation of the resected specimen revealed features of a high-grade tumor comprising of biphasic population of malignant cells. Tumor cells were highly pleomorphic comprising of malignant glial cells including many bizarre tumor giant cells intermixed with bundles of malignant spindle cells. Mitosis was brisk. There was intense endothelial cell proliferation and large areas of palisaded necrosis. Tumor cells showed immunoreactivity for glial fibrillary acidic protein (GFAP) and vimentin [Figure –]. The patient had a prolonged post-op course due to venous edema which gradually resolved, and he has been subjected to radiotherapy.


A 52-year-old woman with faithful and mild personality who has never had medical or psychiatric history and drug/alcohol abuse and dependence was admitted to orthopedic hospital for neck pain after a traffic accident. She was diagnosed with contusion of neck and received painkillers. Three days after the traffic accident, she developed delusions, visual and auditory hallucinations, and her speech became incoherent. Although she was voluntarily discharged from the hospital after 5 days of admission, hallucinations and delusions lasted and she became agitated and violent. She was admitted to the psychiatric ward of our hospital due to psychotic state with disorientation and agitation, 6 days after the traffic accident. On admission (day 1), her physical and neurologic examinations (brain computed tomography, usual blood tests including levels of serum electrolytes, fasting blood glucose, and renal and liver function) showed no remarkable findings. But considerable changes were observed in the plasma levels of HVA and MHPG on admission; the level of HVA was 66.5 ng/mL (normal range for 50–60-year-old subjects, 4.0–15 ng/mL) and the level of MHPG level was 41.8 ng/mL (normal range, 4.0–7.0 ng/mL). Brain magnetic resonance imaging and whole-body computed tomography, electrocardiogram, and chest X-ray were all normal. Thyroid stimulating hormone and free T3 were normal, but free T4 was slightly elevated with 1.79 ng/mL (normal range, thyroid stimulating hormone: 0.50–5.00 μIU/mL; free T3: 2.30–4.00 pg/mL; free T4: 0.90–1.70 ng/mL). Thyroid autoantibodies tests indicated normal titrates of antithyroid peroxidase antibodies 7.7 IU/mL (normal range, 0.0–15.9 IU/mL), but elevation in titrates of antithyroglobulin antibodies (anti-TG) 92.3 IU/mL (normal range, 0.0–27.9 IU/mL).
The clinical course after admission is summarized in . A diagnosis of HE was suspected because of the extensive negative work-up, disturbance of consciousness and psychotic symptoms, and positive anti-TG. On day 16, oral prednisolone (PSL) 50 mg/day was administered. Disorientation, hallucinations, and delusions dramatically improved after the administration of PSL, and plasma levels of HVA and MHPG decreased to 7.2 and 9.9 ng/mL, respectively, on day 19. On day 27, hallucinations and delusions appeared again, and plasma levels of HVA and MHPG increased to 33.1 and 22.4 ng/mL, respectively, on day 33. After that, since a diagnosis of steroid psychosis was suspected, the dosage of PSL was decreased to 45 mg/day and risperidone was started at 2 mg/day. Subsequently, her hallucinations and delusions were alleviated. PSL was tapered, and her psychiatric symptoms gradually improved. On day 82, plasma levels of HVA and MHPG were 17.9 and 7.7 ng/mL, respectively.


A 66-year-old woman developed pain in her left shoulder and was diagnosed with pathological fracture. Her serum IgG was elevated to 3811 mg/dL and IgG-lambda type M-protein was detected by serum immunoelectrophoresis assay. She had 19.6% of bone marrow plasmacytosis with normal cytogenetics. The diagnosis of ISS stage 1 IgG-lambda type MM was established. No other bone lesion, anemia or kidney injury was found. Her left upper extremity was treated with 8 Gy single fraction using a 4MV photon beam by parallel opposed portals, prior to the conventional vincristine/adriamycin/dexamethasone (VAD) induction therapy. After the three courses of VAD treatment, she was switched to a bortezomib/dexamethasone (BD) regimen because of a Helicobacter cinaedi bacteremia developed in the third course of VAD. After the completion of 3 courses of BD regimen, her bone marrow plasmacytes was decreased to 0.4%, however, a tumor of 1×1 cm large developed in her left arm. With a clinical diagnosis of plasmacytoma, second course of radiotherapy with 8 Gy irradiation (left upper extremity was treated with 8 Gy single fraction using a 4MV photon beam by parallel opposed portals), was performed, after which the patient underwent lenalidomide/dexamethasone therapy. Lenalidomide/dexamethasone was effective, and her EM nodule decreased in size to visually undetectable level; however, 6 months later, after 4 courses of Lenalidomide/dexamethasone treatment the nodule enlarged again. There were 15×15 mm tumor in flexor side of her left arm and 20×20 mm tumor in her extensor side of forearm. MRI revealed those tumors were not connected to cortical bones, in addition, no other tumors in her left arm were found. A needle biopsy of a tumor in her left arm was performed and an accumulation of atypical plasmacytes were detected. As a third line therapy, 2 courses of bortezomib/cyclophosphamide/dexamethasone regimen, followed by melphalan/thalidomide/prednisolone (MPT) was administered, however, during the 6th course of MPT, the extramedullary plasmacytoma occupied her left upper limb.
Then, pomalidomide/dexamethasone as a fourth line was started, which was initially effective; the size of the tumor mass decreased, and the vessels on the surface of the bulk of the tumor appeared to be reduced. However, in the 3rd course of pomalidomide treatment, the EMD enlarged again and extended to her left forearm and back of the hand (). On the contrary, there were only scarce MM cells in the bone marrow (3.6%). Those MM cells were morphologically plasmablastic and harbored complex cytogenetic abnormality. She died of severe respiratory failure. Pleural and pulmonary tumor infiltration was suspected. Post-mortem examination revealed extensive MM involvement of multiple organs, including not only the left upper limb, but also lung, liver, kidney, stomach, and thyroid (). However, the bone marrow had only scattered patchy myeloma cells.


A 38-year-old female patient had a past medical history of cervical intraepithelial neoplasia treated with cervical conisation, and a pheochromocytoma that was laparoscopically resected in 2011. She was followed up with yearly magnetic resonance imaging (MRI) that demonstrated a segment 1 liver tumor whose size increased of 20 mm in two years. This 50-mm lesion bore the MRI features of HCA, showing a heterogeneous signal intensity on T-2 weighted images and low-signal intensity on T-1 weighted images. The lesion was slowly and gradually enhanced after injection of gadolinium without significant wash-out on portal phase (Figure ). In addition, a left renal cyst was noticed, described as type 3 according to the Bosniak classification. An 18FDG PET-CT (Figure ) was performed to further confirm the nature of the hepatic lesion and exclude extrahepatic metastases. The liver lesion appeared hypermetabolic with a standardized uptake value (SUVmax) of 9.3. A percutaneous biopsy was performed and immunohistology allowed the diagnosis of I-HCA. Blood carcinoembryonic antigen, carbohydrate antigen 19.9 and alphafoetoprotein were negative. A discussion in a multi-disciplinary oncological team meeting led to the decision of the resection of the hepatic lesion. A laparoscopic resection of hepatic segment 1 was performed, extended to segments 2 and 3 due to the location of the tumor at the junction between the inferior vena cava, the left and middle hepatic veins and the left branch of the portal vein. During the same anesthesia, the left kidney mass was resected through a lombotomy, following the preferences of the urologist. The surgical specimen was analyzed and showed slightly clarified hepatocytes scattered throughout the lesion, fibrous tracts with vascular structures within, probably arteries with thick walls (Figure ). Some inflammatory components surrounded these arteries and there was no significant sinusoidal dilatation. At immunohistochemistry, serum amyloid A was negative and anti-C reactive protein antibodies showed a significant expression of the inflammatory protein around blood vessels, confirming I-HCA (Figure ). Inflammatory cells were CD3 positive (Figure ). The immediate post-operative state was excellent, without significant pain and fast oral feeding. The length of hospital stay was 5 d. The patient was seen again one month later for an evaluation visit and no particular problems were observed.


Our case is a 21-year-old male born with clinical manifestations consistent with Opitz G/BBB syndrome including severe bilateral cleft lip and palate, hypertelorism, low-set prominent ears and midfacial hypoplasia. His history was complicated with respiratory distress requiring tracheostomy at 8 months of age. He had also developed gastric volvulus requiring partial gastric resection and gastrostomy.
In Fig. , the asymmetries at the upper lip, widened and short philtrum, bilateral widened and displaced alar base, poor nose tip projection and shortened columella are shown.
Functional evaluation of the nose revealed a positive Cottle maneuver for bilateral internal valve collapse, external valve collapse on forced inspiration, bilateral retracted and notched ala, shortened columella and poor tip projection.
The patient was planned for a single staged procedure including an open rhinoplasty, complete lip revision and Keen approach to the midface. The surgical plan was devised to address each functional impairment of the craniofacial defects as shown in Fig. . The procedures were planned with the following sequence:
Bilateral internal nasal valve collapse with bilateral spreader grafts Bilateral external nasal valve collapse with bilateral batten grafts Bilateral retracted and notched ala with bilateral rim grafts Shortened columella with double V–Y flaps Poor tip projection with columellar strut Hypoplastic midface and pyriform rim with costocondral rib grafts Lip shortening and vermillion deficiency with dermal graft
A 6-month follow-up was scheduled to review the restorative results. Surgical correction of the severe cleft lip, nasal deformities and midfacial hypoplasia showed significant improvement in symmetry and aesthetic result. Nasal airway function was optimal at both the internal and external nasal valves as evidenced by negative Cottle maneuver. The alar base was corrected to a symmetric position. The grafts maintained their structure and had little resorption. Bone grafts proved to provide adequate scaffolds at the pyriform rim. The patient has not undergone further revision.


A 23-year-old male with no known comorbidities presented with complaints of vague pain in his epigastrium with frequent bouts of bilious vomiting for 10 days. Clinical evaluation revealed no abnormality except for vague tenderness to deep palpation in the right hypochondrium and epigastrium. The routine laboratory tests were unremarkable. Ultrasound of the abdomen revealed a hypoechoic lesion around 30 × 20 mm in the region of the porta hepatis with a contracted gall bladder (GB) and no intra-hepatic biliary radicle dilatation (IHBRD). Magnetic resonance cholangiopancreatography (MRCP) revealed a saccular mass in relation to the cystic duct close to its insertion into the common hepatic duct (CHD), with no IHBRD (Fig. ). The right and left biliary systems were normal with no abnormality detected in the pancreatobiliary junction. A provisional diagnosis of Type II choledochal cyst/double GB/Type VI choledochal cyst was made.
We planned to do a diagnostic laparoscopy with cholecystectomy +/− common bile duct (CBD) excision with hepaticojejunostomy. Laparoscopy was performed using the conventional four-port technique. The dissection commenced by removing adhesions between the cystic structure and the surrounding omentum and duodenal loop (Fig. ). The cyst was dissected free from the surrounding structures and the insertion of cystic duct into the CHD was delineated (Fig. ). There was no communication between the cyst and the CBD. Therefore, a decision to do a cholecystectomy alone was made. The GB was contracted. It was dissected and removed en masse with the cystic mass. The post-operative course was uneventful and he was discharged in stable condition on Day 3 after surgery.
The gross specimen revealed a contracted GB with an isolated mass attached to the anterolateral wall of the cystic duct. The specimen measured 6.5 cm in length, out of which GB was 3.5 cm in length and 3 cm in diameter (Fig. ). Instilling saline into the GB caused the cystic mass to fill up suggesting a communication with the GB lumen. Microscopy showed papillary hyperplasia of the mucosa with features of chronic inflammation in the wall of the GB.


The patient was a 25-year-old male mine worker, who suffered a multiorgan injury when he was buried by falling rocks. On admission to the Intensive Care Unit, he was in a severe condition and exhibited symptoms of cardiopulmonary failure. The patient was intubated – mechanical ventilation was introduced along with pharmacological therapy with catecholamines and antibiotics. After a chest radiogram revealed right-sided pneumothorax and costal fractures (ribs II–V), a drain was introduced into the right pleura. The patient’s crushed right foot was treated surgically (amputation at Chopart’s joint), while the fractured medial condyle of the femur was treated conservatively. Due to the observed incidents of reduced respiratory fitness parameters, computed tomography of the chest was performed on the 3rd day after the trauma. The examination visualized irregularities in the outline of the left main bronchus and a consolidation of gas bubbles in this area, which prompted the suspicion of bronchial injury (, present a tomographic reconstruction of the respiratory tract).
Diagnostic investigation was supplemented with bronchofiberoscopic examination, revealing hyperemia and massive edema of the mucosa in the proximal segment of the left main bronchus, immediately next to the tracheal bifurcation. Consequently, the attempt to reach the left main bronchus with the endoscope was unsuccessful.
The patient was referred for further treatment at the Thoracic Surgery Center in Wrocław with a suspicion of airway disruption. A decision was made to perform urgent surgical treatment. Left posterolateral thoracotomy was performed. Typical post-traumatic lesions were found intraoperatively: a pleural hematoma (approx. 500 ml in volume) and extensive hematomas in the tissues of the thoracic integuments. A small amount of air was found in the mediastinum. Prepare of the mediastinal tissues revealed the completely amputated left main bronchus; the whole length of the membranous part of the bronchus was ruptured. The bronchial membrane was sutured longitudinally. Subsequently the detached bronchus was anastomosed with the trachea, and the anastomosis was wrapped with an intercostal muscle flap.
The patient’s hospitalization continued at the Intensive Care Unit of the Lower Silesian Lung Diseases Center. The dosage of catecholamines and sedatives was gradually reduced. On the 3rd day, the drain from the left pleura was removed; 3 days later, the right pleural drain was removed as well. On the 6th day after the thoracotomy, the airway was reevaluated with an endoscope, revealing stenosis at the tracheobronchial anastomosis; the endoscope could not pass through this location. Additionally, a large amount of mucous secretion, which filled the whole bronchial tree, was evacuated. A decision was made to wake and extubate the patient. Consequently, the secretion accumulated again, resulting in a repeated bronchoaspiration after less than 20 h. It was deemed necessary to perform tracheotomy. After the patient’s cardiopulmonary parameters stabilized on the 14th postoperative day, the patient was referred for further treatment on the Thoracic Surgery Department. The treatment consisted in rehabilitation and fitness improvement. The patient was discharged from the Center on the 18th postoperative day. After less than 6 weeks, the patient was readmitted with symptoms of left main bronchus occlusion. For the past several days, the patient had manifested symptoms of airway infection: he was fever and he had dyspnea. An out-patient computed tomography (CT) scan demonstrated anastomotic stenosis (), but lung aeration was maintained ().
A chest radiogram () taken on the day of readmission (7 days after the CT exam) revealed complete atelectasis of the left lung.
Bronchofiberoscopic examination showed that the lumen of the left main bronchus was very narrow: its diameter did not exceed 3 mm. An attempt was made to perform mechanical dilatation, but without success.
The diagnostic investigation was supplemented with chest CT, revealing obturation of the left bronchial tree () and a complete lung atelectasis (–).
A decision was made to perform surgical intervention. Thoracotomy was repeated with sleeve resection of the stenosed left main bronchus (length: 1–1.5 cm), and the bronchus was anastomosed with the trachea (the posterior wall with single sutures, and the anterior wall with a continuous suture). shows the chest radiogram obtained on the 1st postoperative day, while presents a radiogram from the 6th postoperative day.
From the day of readmission, the patient was administered broad-spectrum antibiotic treatment. On the third postoperative day, the drain placed in the left pleura was removed. The patient was discharged on the 7th postoperative day. He continued to be monitored as an outpatient – he attended follow-up visits, during which the site of anastomosis was evaluated with an endoscope. The first such examination took place 2 weeks after discharge, demonstrating permeability of the anastomotic site and no signs of inflammation. Subsequent follow-up examinations took place every three months until the end of February, 2015. The final examination showed a stable endoscopic image with satisfactory width of the left main bronchial lumen.


A 22-year-old male, driver of the car was involved in a road traffic accident when his car hit against a bridge with his ipsilateral hip and knee in flexion. He presented to the emergency department in 4 h injury time with painful deformities of the right hip and knee, along with Type I open wound right leg with abnormal mobility suggestive of fracture in the ipsilateral leg. On examination, he was drowsy and disoriented with a Glasgow Coma Scale 8/15 and found to be hemodynamically unstable. Physical examination of the extremities revealed a flexed and adducted right hip with the right knee flexed to 30° and with posterior dislocation (). Neurovascular examination revealed common peroneal nerve palsy and absent distal pulses. However, the Doppler showed weak signals over posterior tibial and dorsalis pedis artery, and the toes were found to be warm with delayed capillary filling. Computed tomography (CT) brain showed acute hemorrhagic contusion. Radiographs showed a posterior dislocation of the right hip and posterior dislocation of right knee with comminuted fracture right distal tibia ().
The patient was intubated and hemodynamically stabilized and immediate closed reduction was attempted. The Firstly, an ankle spanning exfix was applied to address the distal tibial fracture, and the knee dislocation was reduced by gentle traction given to the leg. Further traction on the right hip with the hip in 90° flexion with an assistant stabilizing the pelvis reduced the right hip dislocation, which was found to be stable after reduction. Then, ankle spanning external fixator extended to span the knee. Immediately following the procedure, distal pulses were clinically not palpable but relatively good flow was noted in the Doppler both in the posterior tibial and dorsalis pedis artery. The foot continued to be warm with good capillary filling of the toes. The patient was on continued ventilator support for 48 h in Intensive Care Unit and serial monitoring of the right lower limb vascularity was done using Doppler. Post reduction CT of the pelvis showed only a minor posterior wall fracture with congruently reduced hip joint () allowing conservative management. Distal pulses were felt clinically from 4th day following which locking compression plate fixation of distal tibia was done. The patient general condition improved and discharged after 3 weeks.
At 6 weeks, external fixator was removed and bone grafting was done for the distal tibial fracture and the right knee gently manipulated. The distal tibial fracture healed in 4 months with good recovery of common peroneal nerve. The knee flexion was 0-130° with posterior sagging and multidirectional instability. Magnetic resonance imaging (MRI) of right knee showed posterior cruciate ligament (PCL) avulsion fracture and lateral collateral ligament avulsion fracture (). It was planned arthroscopic evaluation which showed a torn PCL at tibial attachment and torn anterior cruciate ligament (ACL) at anterolateral bundle but the continuity maintained with posterolateral bundle. Arthroscopic PCL reconstruction using bio screws and open posterolateral corner repair using hamstring graft and endobutton was done. Post-operative rehabilitation included the protocol for multi-ligament instability. At 2 years follow-up, he had full range of movements at the right hip and knee. Radiographs of the pelvis showed a viable femoral head without signs of avascular necrosis. There was complete recovery of the common peroneal nerve (, ). The knee was stable and he returned to his preinjury level of activity.


A 45-year-old female was referred for evaluation of an asymptomatic nodule on the lower lip of 4-week duration. Clinical examination revealed a well-defined, nontender, ulcerated, nodular growth measuring approximately 1 cm × 1 cm on the lower lip vermilion []. Oral hygiene was fair. There was no report of local trauma, and the patient presented no parafunctional habits. The patients’ medical, family and social history was noncontributory.
An excisional biopsy was performed under local anesthesia. Histopathological examination of H- and E-stained section revealed a hyperkeratotic, hyperplastic epithelium with parakeratin plugging in some areas. The underlying connective tissue stroma showed multiple areas of epithelial tissue entrapment with cystic degeneration in the center and keratinization [Figures and ]. Periodic acid–Schiff (PAS)-stained section showed glycogen granules in the superficial epithelium. The epithelial proliferations in the connective tissue did not exhibit PAS positive glycogen granules though [].
The lesion healed with no scarring over 5 weeks, but the patient was lost to follow-up later.


A 26-year-old male patient with intractable head ache and recent onset focal seizures came to our department after initial evaluation done elsewhere. His neurological examination revealed nothing remarkably. Enhanced computed tomography (CT) and magnetic resonance (MR) evaluation showed lesion with patchy calcification and patchy enhancement in left frontotemporal region with possible dural attachment, which made us to make a preoperative diagnosis of meningioma [Figures and ].
Patient underwent total surgical excision of the lesion along with involved dura. The lesion was hard and avascular in most of the areas with clear plane between the brain parenchyma except for the area of dural attachment [].
Duraplasty was done with pericranium. Postoperatively, patient developed focal seizures with generalization and postictal confusion, weakness of right side limbs and aphasia. He was treated with escalated doses of two antiepileptic drugs (AEDs). Postoperative CT scans showed complete tumor excision []. Patient recovered from his postictal deficits completely and discharged with normal neurology. Pathological examination revealed chondromatous elements, which confirmed nature of the lesion as chondroma []. Patient attending follow-up and on the course of tapering of AEDs.


A 24-year-old woman with keloid scars on her upper back had been treated with multiple intralesional injections of triamcinolone acetonide plus 1% lidocaine (xylocaine) during the previous 10 years without any reaction. After her most recent treatment, she developed generalized urticaria, lip angioedema, numbness on her face, abdominal pain, vomiting, chest tightness, and hypotension 2 minutes after injection. Lidocaine hypersensitivity was suspected. She decided to stop the treatment, without any medication being prescribed for her condition. She was healthy, with no history of any other medication use and no history of drug/food allergy or atopic diseases. After 1 year she returned for intralesional injection of triamcinolone acetonide only (without lidocaine), and was scheduled once a month for this regimen. The first injection was tolerated well. The reaction occurred at the time of the second injection, with numbness on her face developing 15 minutes after injection and 5 minutes later with urticaria on her chest wall and both arms, together with hypotension (blood pressure of 90/60 mmHg). At the emergency room, her vital signs revealed blood pressure 90/60 mmHg, pulse rate 72 beats/min, respiratory rate 20 breaths/min, temperature 37.2℃, and oxygen saturation 95% on room air. Immediate treatment with an intramuscular injection of adrenaline, intravenous dexamethasone and chlorpheniramine was given, and the patient quickly recovered. Laboratory investigation showed white blood cells 13.7×109/L (neutrophils 86%, lymphocytes 11%, monocytes 2%, eosinophils 1%) and serum tryptase 5.89 ng/mL (1.9–13.5 ng/mL).
Anaphylaxis was diagnosed. The allergology workup was performed 4 weeks after the anaphylaxis event to clarify the identity of the culprit drug. Skin tests with triamcinolone acetonide, lidocaine, and the excipients carboxymethylcellulose (CMC), benzyl alcohol and polysorbate 80, were performed, together with alternative corticosteroids (hydrocortisone, methylprednisolone, and dexamethasone).
The skin test concentrations were as previously reported [] and are summarized in . Histamine and normal saline were used as positive and negative controls, respectively. The skin prick test (SPT) was considered positive when a wheal of more than 3 mm in diameter presented after 15 minutes. If the SPT was negative, an intradermal test (IDT) was performed by injecting 0.02–0.05 mL of the reagent solution intradermally. Results were interpreted after 20 minutes, and a positive result was declared when the wheal and erythema extended more than 2 mm from the initial injection papule []. When SPT and IDT were negative, a provocation test was performed.
The patient had a positive SPT for triamcinolone acetonide (), which confirmed IgE-mediated hypersensitivity (). The SPT for the other corticosteroids, including hydrocortisone, methylprednisolone, and dexamethasone, were negative. Subcutaneous provocation with lidocaine (cumulative dose of 3.2 mL of 1% lidocaine) and oral CMC (cumulative dose of 250 mg) was well-tolerated and considered negative.


A 70-year-old woman, affected by anxious-depressive syndrome in antidepressant and antipsychotic therapy, was referred to our hospital ward for a syncope occurring during vomiting and diarrhea. In the emergency room, the electrocardiogram (ECG) showed a junctional rhythm, and the echocardiography was normal. During the cardiology stay, ECG monitoring recorded a sinus bradycardia with significant pauses, 6 s long [].
Next day, a bicameral pacemaker (PMK) was implanted. The patient came back to the cardiology ward after PMK implantation, and ECG showed activity PMK-induced [].
A few minutes later, the woman had an acute episode of detachment from reality and generalized muscle spasms, considered first as a comital crisis, and treated with intravenous midazolam. We immediately required a psychiatry advice; an accurate psychiatry evaluation made diagnosis of convulsive hysteric crisis.
Next day, ECG showed activity PMK-induced with significant persistent anterior (V2–V6) ST-segment elevation []. Troponin levels were elevated (2.74 ng/ml, the cutoff is 0.04 ng/ml). Echocardiography showed left ventricular apical and midventricular segments - akinesis, basal segments - hyperkinesis, and reduced left ventricular ejection fraction [].
We decided for an urgent coronary angiography to exclude an acute coronary syndrome; it showed the absence of obstructive coronary disease. Ventriculography confirmed our ECG suspicion demonstrating left ventricular apical ballooning, a typical feature of TC [].
In the following days, ECG [] and troponin levels came back normal; serial ECG assessments revealed progressive recovery of systolic left ventricular function, first midventricular segments recovered a normal contractility and then apical segments too.
Diagnosis of TC was sure, meeting every criterion proposed by Mayo Clinic.[]
The patient kept continuous disorientation, narrowing of the state of consciousness; she never reported chest pain or dyspnea; a lot of other pseudoseizures occurred.
Treatment was mainly guided by psychiatric disease using sedatives, antidepressant, and antipsychotic drugs; cardiologic therapy was occasionally administered, with difficulty.


A 43-year-old, right-handed man without a prior history of epilepsy presented to his neurologist for evaluation of a GTC seizure. He had no other symptoms and was neurologically intact. He was prescribed an antiepileptic drug and underwent radiological evaluation.
Magnetic resonance imaging (MRI) of the brain revealed a nonenhancing lesion measuring 1.5 × 1.8 cm in the right mesial temporal lobe with minimal mass effect. The lesion appeared hyperintense on T2-weighted images [Figure and ]. Fluid-attenuated inversion recovery (FLAIR) sequences delineated a nonspecific hyperintense focus in the right temporal lobe near the gray-white interface [Figure and ]. Diffusion-weighted imaging (DWI, images unavailable) did not reveal any abnormal restricted diffusion. Positron emission tomography (PET, not shown) was obtained, but did not reveal significant uptake of 6-[18F] Fluoro-L-DOPA (18F-FDOPA) in the right mesial temporal lobe. The radiologic features of the lesion were suggestive of a low-grade neoplasm, and the patient was referred to neurosurgery.
A right pteronial craniotomy was performed for resection of the mass. The lesion was noted to have a firm consistency, ill-defined planes, and significant pial adhesions. The lesion was meticulously resected under the operative microscope, and the specimen was sent to pathologic examination. The patient tolerated the procedure well. Immediate postoperative imaging demonstrated confirmed gross-total resection without evidence of ischemia or abnormal enhancement. At 2-week follow-up, the patient was free from seizures and no longer required antiepileptic drugs. The patient has not returned in the 13 months since the surgery.
Histological examination of the lesion revealed normal gray matter with focally prominent polyglucosan body accumulations in the brain parenchyma. Numerous spherical polyglucosan bodies were identified on Hematoxylin and Eosin (H and E) frozen section slides without features of neoplasia. Permanent section H and E slides [] also demonstrated the presence of numerous polyglucosan bodies, far in excess of those found in the normal aging brain. Periodic-acid Schiff stain highlighted the polyglucosan bodies [], providing a final diagnosis of a dense CA lesion. This retrospective review of a single patient's medical record did not require informed consent by the Institutional Review Board. Nevertheless, all images are completely de-identified.


A 56-year-old male patient treated on a clinical trial for bronchopulmonary carcinoid tumors metastatic to the orbit was hospitalized due to complaints of polyuria, polydipsia, weakness, and visual changes. He had experienced polyuria and polydipsia for the past few weeks, and weakness and visual changes over the past few days. One month prior he had been treated for hypertensive urgency with lisinopril and Toprol XL. No previous history of type 2 diabetes mellitus was present. His recent oncology treatment history was significant for focal irradiation of orbital metastases. On admission, his blood pressure was 134/70 mm Hg with a heart rate of 97 beats/min. The rest of the physical examination revealed dry skin, moon face appearance with red cheeks, purple-colored striae on the skin, muscle wasting, and muscle weakness, which was suspicious for Cushing syndrome. A buffalo hump was not present (Fig. ).
His laboratory examination results were notable for hyperglycemia (venous blood glucose 615 mg/dL) and metabolic alkalosis (pH 7.54; bicarbonate 32.5 mmol/L [normal: 22–26]) without ketonuria. Other values were as follows: glycated hemoglobin (HbA1c) 9.7%; potassium 2.7 mmol/L (normal: 3.5–5.1); sodium 149 mmol/L (normal: 136–145); lactate 4.9 mmol/L (normal: 0.5–2.2), and blood urea nitrogen 23 mg/dL (normal: 7–20). Blood cultures and urinalysis were negative for evidence of infection. No EKG abnormalities were present.
Based on these clinical and laboratory findings, the patient was diagnosed with HHNS. Following intravenous hydration, potassium replacement, and regular insulin infusion (0.1 IU/kg/h), the patient reported immediate symptomatic improvement. On suspicion of Cushing syndrome, an endocrine workup was started, which included measurement of serum cortisol and plasma ACTH. The values were as follows: serum cortisol 40.5 mg/dL (8: 00 a.m.; normal: 5–25) and plasma ACTH 112.3 pg/mL (normal: 7.2–63.3), confirming the diagnosis of Cushing syndrome. It is unclear whether the emergence of Cushing syndrome represents active secretion of ACTH from the tumor due to disease progression, although a recent CT scan showed stable disease with central tumor necrosis. A more intriguing possibility is that the ACTH contained within the tumor was passively released, i.e., liberated into the circulation from dead or dying neuroendocrine cells due to recent irradiation of the patient's orbital metastases.


A 33-year-old woman presented at the emergency department with a subacute progressive bilateral leg weakness. The neurological examination revealed a severe paraparesis, as well as hypesthesia of both lower limbs in the absence of sphincter incontinence or radicular pain. Reflexes were hyperactive in both legs, with no evidence of clonus or Babinski sign. The patient had been diagnosed 13 months before with a temporal lobe GBM (Fig. ). She had undergone surgical gross total tumor resection and postoperative MRI scans had revealed a very small residual contrast-enhancing abnormality (Fig. ). Postoperatively, the patient had no other remarkable neurological deficits besides a light remaining right hemiparesis and had undergone concomitant radio- and chemotherapy followed by 6 cycles of adjuvant temozolomide. Nevertheless, the doses of temozolomide had to be reduced due to a severe thrombocytopenia developed by the patient during the treatment.
Brain MRI at the time of consultation due to paraparesis showed no evidence of a local intracranial recurrence (Fig. ). However, MRI of the spine demonstrated a homogeneous contrast-enhancing mass within the spinal canal at the Th8/9 level causing significant spinal cord compression (Fig. ). A lumbar puncture was performed and cytological examination of the cerebrospinal fluid (CSF) did not reveal the presence of neoplastic cells.
Due to the spinal cord compression and progressive paraparesis, we suggested surgery with the aim of decompressing the spinal cord and if possible remove the tumor. Therefore, we performed a decompressive laminectomy at the Th8/9 level. After opening the dural sack, an intradural intramedullary high vascularized infiltrating tumor was identified (Fig. ). Due to its adherent and infiltrating pattern with no recognizable edges to the normal tissue, it was unreasonable to attempt a radical tumor excision so we carefully performed a biopsy and interposed a dural graft to expand the spinal canal, therefore reducing the pressure against the spinal cord (Fig. ).
Unfortunately, neurological deficits did not improve during the postoperative follow-up and the patient refused to undergo further oncologic therapies. She underwent best supportive care and died 5 months later due to complications of pneumonia acquired in the context of a highly debilitated general state of health.