We present the case of a 44-year-old woman who attended the antenatal care clinic after six weeks of amenorrhea. The pregnancy was confirmed by positive urine β-hCG testing. She did not have any vaginal bleeding. She had four live births by low-transverse cesarean section with no previous miscarriages or ectopic pregnancies. The first two cesarean section deliveries were due to failure to progress. Her last pregnancy was four years ago. The past medical history revealed ulcerative colitis managed by mesalamine.
On examination, she had a normal blood pressure of 125/76 mmHg, a pulse rate of 89 beats/min and body temperature of 37.1 °C. Her abdomen was soft, lax and non-tender. Her cardiorespiratory and neurological systems were normal. The results of pelvic examination were normal. Laboratory investigations revealed hemoglobin level of 12.7 g/dL and β-hCG level of 93,788 mIU/mL. The patient underwent transvaginal ultrasound examination which identified the gestational sac with fetal pole and cardiac activity located in the anterior part of the lower uterine segment near the cervicouterine junction along with empty uterine cavity (Figure ). The crown-rump length of the embryo was 2.10 cm, consistent with gestational age of eight weeks. On Doppler examination, hyperechoic rim of choriodecidual reaction with prominent vascularity was noted. As the findings were suggestive of cesarean section scar ectopic pregnancy, the patient underwent MRI scan which confirmed the diagnosis of ectopic scar pregnancy (Figure ). The MRI findings demonstrated a gestational sac measuring 3.5 × 2.9 × 3.5 cm implanted in the anterior wall of the lower uterine segment in the region of the scar of previous cesarean section. The gestational sac had T1-isointense and T2-hypointense fetal pole and was surrounded by decidual reaction. Posteriorly, the gestational sac was communicating with the endometrial cavity.
The patient was counselled regarding management options of ectopic pregnancy including medical and surgical treatment with thorough explanations of advantages and disadvantages of each choice. She chose medical treatment because of her strong desire to preserve her uterus and maintain future fertility. Therefore, she received an 80 mg of methotrexate into the ectopic gestational sac under transvaginal ultrasound-guided technique. There was no internal bleeding or lower abdominal pain noticed following the procedure. The patient was admitted to the ward for observation. A repeat serum quantitative β-hCG after two days from the injection revealed a level of 50,057 mIU/ml, showing 46% drop from the initial result. Four days following the procedure, the patient complained of lower abdominal pain and profuse vaginal bleeding. On physical examination, lower abdominal tenderness without rigidity or guarding was noted. Laboratory investigations revealed a 4.9 g/dL drop in her hemoglobin level.
Urgent abdominal computed tomography scan was performed and it demonstrated a bulky uterus with a cystic structure that was surrounded by hyperdense attenuation at the junction of middle and lower thirds of the uterus along with the presence of high volume of high-density fluid in the abdominopelvic cavity (Figure ). These findings indicated hemoperitoneum due to a ruptured ectopic scar pregnancy. Therefore, the patient was resuscitated and prepared for emergency laparotomy for excision of the ectopic scar pregnancy with possible hysterectomy. During surgical exploration, a soft mass was seen at the site of previous cesarean scar. An incision was made over the mass and the products of conception were removed. The bulge was noted to be communicating with the uterine cavity. The edges of the scar tissue were excised and freshened. The obtained tissue was sent for histopathological examination which revealed the presence of products of conception with phenotypically male fetus of seven weeks of age. The patient tolerated the procedure well and had uneventful recovery. The serum β-hCG was 10,894 mIU/ml on the first post-operative day. She was discharged on the fifth post-operative day in a stable condition. The patient had weekly clinical evaluation and measurement of serum β-hCG level. She was asymptomatic and had no active complaints. Her serum β-hCG was undetectable on the 35th day after the methotrexate injection.


A 46-year-old Swahili speaking African female presented to the emergency department with complaints of sudden shortness of air (SOA) for three days. SOA worsened with exertion and was associated with headache, back pain, and vague chest discomfort. She had a past medical history of hypertension treated with lisinopril and propranolol. She had emigrated to the United States within the last two years.
On physical examination, her vital signs were as follows: oral temperature, 98.4 Fahrenheit; heart rate, 98 beats per minute (bpm); respiratory rate, 28 breaths per minute; and blood pressure, 158/97 mmHg. Neck examination did not show lymphadenopathy or thyromegaly but she complained of pain on palpation in the submandibular area. Cardiac examination showed rhythmic heart sounds, regular and without murmur; lungs were clear to auscultation, but she had labored breathing and tachypnea. Abdominal examination showed mild tenderness on palpation of the right upper quadrant, no distention, and regular bowel sounds. The rest of the examination was unremarkable.
A computed tomography angiogram (CTA) of the chest (Omnipaque 350, 100 mL IV contrast = 35 million mcg of organic iodide) ordered in the emergency department was negative for pulmonary embolism, but it showed an incidental 1.8 cm left thyroid nodule (Figure ). Four hours later when she was evaluated by the admitting team, TSH level ordered was 0.40 uIU/mL. No free triiodothyronine (FT3) and free thyroxine (FT4) levels were ordered at this time, but they were added later (Table ). Hematology workup was relevant for a white blood cell count of 16.6 x 103/cmm.
Later that night, the patient became more tachycardic with a heart rate of 110 bpm and respiratory rate of 30 breaths per minute. Electrocardiogram done at the time showed sinus tachycardia (Figure ). TSH, FT3, and FT4 levels were ordered with the following results: 0.27 uIU/mL, 12.4 pg/mL, and 2.67 ng/mL, respectively. It was decided to start the patient on methimazole and prednisone, uptitrate propranolol dose, and test for thyroid-stimulating immunoglobin (TSI) and thyroid peroxidase (TPO) antibodies due to suspicion of thyroid storm. Endocrinology was consulted the next day, and it was decided to stop methimazole and prednisone two days after initiation as it was thought that hyperthyroidism was precipitated by iodine contrast load received during CTA. FT3 and FT4 levels were followed during the remainder of her hospitalization with normalization of values (Table ). TSI and TPO antibodies were normal, supporting the hypothesis of IIH. Thyroid ultrasound showed a single right thyroid nodule measuring 6 mm and two left thyroid nodules measuring 15 and 19 mm, respectively. There was no increased vascularity of the thyroid, which would be suggestive of Graves’ disease. Thyroid scintigraphy was not pursued as the patient had received methimazole recently.
Further investigation for patient’s leukocytosis revealed a peritonsillar abscess. Once antibiotic therapy was initiated, the patient gradually returned to normal.
The patient had repeat thyroid function testing nine weeks after the initial presentation with normalization of TSH and FT4, confirming the diagnosis of transient hyperthyroidism consistent with the Jod-Basedow phenomenon/IIH.


A 61-year-old woman presented with symptoms of intractable nausea and vomiting for 10 days. Her past medical history included permanent hypothyroidism and hypoparathyroidism from subtotal thyroidectomy due to a huge multinodular goiter 2 years earlier. Her thyroid condition was well controlled with levothyroxine 600 μg per week. She was also prescribed with 2,400 mg of elemental calcium and 1 μg of alfacalcidol per day. On the last follow-up, 2 months prior to this visit, she was in good health with normal calcium and thyroid function levels. A thorough medical history review discovered that she had been taking different preparations of over-the-counter alfacalcidol, 0.25 μg to 1 μg/tablet for 6 weeks. She also increased the daily calcium supplement up to 4,800 mg of elemental calcium per day to control her tingling sensation. On physical examination, the patient was drowsy and moderately dehydrated. Her vital signs and neurological examination were unremarkable. Laboratory investigations showed profound hypercalcemia, mild hypophosphatemia and azotemia: serum calcium 17.08 mg/dL (8.5–10.5), phosphate 2.1 mg/dL (2.5–4.5), albumin 3.5 mg/dL and creatinine 2.0 mg/dL. Her venous pH was 7.46 and electrolytes showed mild hypokalemia and metabolic alkalosis: sodium 135 mEq/L potassium 3.4 mEq/L, chloride 95 mEq/L, and bicarbonate 29 mEq/L. Low levels of intact PTH 6.07 pg/mL (15–65), 25-OH vitamin D 19 ng/mL (>20) with a normal level of 1,25(OH)2 vitamin D 25.4 pg/mL (19.9–79.3) and normal levels of PTHrP were shown. Her thyroid function test was normal: FT4 1.35 ng/dL (0.80–1.80) and TSH 2.52 μIU/mL (0.35–4.10). The electrocardiography (ECG) showed sinus rhythm with normal QTc interval (410 ms). Her amylase and lipase levels were not elevated. The presumptive diagnosis of CAS was proposed from her triad of hypercalcemia, metabolic alkalosis, and renal insufficiency. She responded well to intravenous hydration with isotonic normal saline (4 L/d), subcutaneous calcitonin (200 IU q 8 h) and discontinuation of calcium and vitamin D supplements. Her clinical symptoms and serum calcium level returned to normal within 2 days of treatment. The patient was discharged on the 4th day of hospitalization and was prescribed with 2,000 mg of calcium carbonate and 0.5 μg of alfacalcidol daily. During the follow-up after 6 months, she did not have experienced episode of hypercalcemia and her renal function returned to baseline level.


A 55-year-old female with a past medical history of cocaine use and hypertension was admitted for a large right basal ganglia hemorrhage with extension into all four ventricles requiring right decompressive craniotomy. Following a fever on day 23 of hospitalization, a head CT scan was obtained which revealed development of 3 new distinct parenchymal hemorrhages within the prior right cerebral hemorrhages. At this time, the patient's fever prompted initiation of empiric vancomycin and ceftazidime. On day 26 of hospitalization, the parenchymal hemorrhages were managed with neurosurgical debridement of the hemorrhagic brain with partial temporal lobectomy and duraplasty with titanium mesh placement since the patient failed to improve with medical management alone. A repeat head CT scan obtained on day 28 of hospitalization identified an 18 mm subgaleal fluid collection overlying the duraplasty noted in . Given the persistent fevers, the subgaleal fluid collection was aspirated on day 30 of hospitalization which subsequently grew 2+ Enterobacter cloacae complex susceptible to TMP-SMX, gentamicin, ciprofloxacin, and ertapenem. Resistance patterns of the patient-specific E. cloacae isolated are described in . Cell count of the cerebrospinal fluid (CSF) identified 17,300 nucleated cells/μL with 98% granulocytes, 35,000 red cells/μL, 980 mg/dL of protein, and an undetectable glucose level.
Given the susceptibilities, the patient was transitioned from vancomycin and ceftazidime to SMX-TMP (5 mg/kg of TMP) every 8 hours IV and metronidazole 500 mg IV every 12 hours. Since the patient continued to have persistent fevers, a repeat head CT scan was performed which identified an enlarging subgaleal fluid collection measuring 104 × 23 mm as noted in . The neurosurgical team believed the risk of removal of the titanium mesh was much greater than the benefit given her poor surgical candidacy attributed to comorbidities. Furthermore, a future procedure would be needed following removal. Repeat subgaleal collection aspirations performed on day 35 of hospitalization remained persistently positive for E. cloacae prompting placement of a lumbar drain. Daily intrathecal gentamicin at a dose of 4 mg was started at day 36 of hospitalization and was administered daily through the lumbar drain. A follow-up head CT scan was performed and revealed no significant change in the subgaleal abscess size. Given the lack of improvement on imaging, repeat cultures of the subgaleal fluid collections were obtained and remained positive for E. cloacae. Repeat CSF cell count revealed 139,000 nucleated cells/μL with 95% granulocytes, 351,000 red cells/μL, 1,910 mg/dL of protein, and an undetectable glucose level. Given the persistently positive cultures and worsening CSF profile, after 11 days of SMX-TMP and metronidazole, the patient was transitioned to ertapenem 1 g IV daily. A CSF culture obtained 3 days after the initiation of ertapenem was found to be sterile. CSF cell count from this specimen revealed improvement and identified 112 nucleated cells/μL with 83% granulocyte predominance, 7 red cells/μL, 449 mg/dL of protein, and a glucose level of 32 mg/dL. An MRI of the brain obtained 7 days after ertapenem was initiated revealed a decrease in the subgaleal fluid collection from 18 mm to 8 mm as noted in .
On day 50 of hospitalization, intrathecal gentamicin was stopped. The lumbar drain was removed after 2 weeks of therapy with ertapenem given achievement of a drainage rate of less than 10 mL/hour. After 16 days of ertapenem, the patient was transitioned to SMX-TMP for prolonged therapy given the presence of the titanium mesh. She was ultimately discharged to a rehabilitation facility after 77 days of hospitalization.


A 38-year-old man presented to a community hospital complaining of 5 days of gradually worsening headache and fatigue with associated vomiting, neck stiffness, photophobia, sweats, and chills. He had been seen at immediate care and was presumed to have influenza. The morning of presentation, the patient experienced altered mental status and was brought by his family to the emergency department. He was febrile at 39.4°C and tachycardic to 110 BPM with somnolence. Neurological examination was nonfocal, but positive nuchal rigidity and poor dentition were noted. Blood cultures x2 were drawn. Labs were remarkable for elevated fasting glucose of 318, procalcitonin-2.38, and WBC-19. Urinalysis was normal. CT head without contrast showed no abnormalities (). Lumbar puncture CSF studies showed xanthochromia with glucose-133, protein-642, RBCs-295, and nucleated cell count-12,000 with differential: segs-72, monocytes-25, lymphocytes-2.0, basophils-1, and eosinophils-0. The infectious diseases service was consulted on suspicion of bacterial meningitis. CSF viral panel and culture were obtained, and then he was started on empiric vancomycin, ceftriaxone, and dexamethasone as well as one dose of acyclovir. In the subsequent 3 days, he was diagnosed with diabetes mellitus (DM) based on hemoglobin A1c of 10.9, mental status improved significantly, and sepsis subsided. Respiratory and CSF viral panels were negative. CSF culture showed no growth, and West Nile CSF IgM was negative. WBC count downtrended to a normal level of 11 by day 4 of admission, and vancomycin and dexamethasone were discontinued with plans to have PICC placed for 2 weeks of ceftriaxone alone. On day 5, he worsened with headaches, fever to 39.2°C, nausea/vomiting, and worsening leukocytosis with WBC count of 12.1. Vancomycin was restarted. White blood cell count rose to 13.2. Both transthoracic and transesophageal echocardiograms did not find vegetations. MRI brain () and C-spine were ordered to exclude parameningeal focus of infection. This revealed a multiloculated cystic lesion representing a 3 cm abscess centered over the posterior right thalamus with adjacent meningitis. No ventriculitis or ependymitis was appreciated. Chest X-ray was unremarkable. IV.
Metronidazole was added. Neurosurgery was consulted. Stereotactic aspiration procedure was not recommended due to high risk and relatively low chance of microbial diagnosis. Continuing antibiotic therapy was recommended with plans to repeat MRI brain in one week. Repeat CSF showed no growth. By day 9, WBCs normalized to 7.5, and symptoms improved but low-grade fevers around 38.2–38.4°C persisted. Repeat MRI brain one week later showed no improvement. Fevers again climbed as high as 39.6°C, and neurosurgery again determined drainage to be too risky. Ceftriaxone and metronidazole were discontinued. Meropenem was started and vancomycin was continued. His serum glucose became well-controlled with the use of basal bolus and prandial insulin. On day 9, he was transferred to a university hospital where MRI demonstrated increasing reach of the infectious material to the posterior third ventricle and into the contralateral thalamus, and again no ventriculitis or ependymitis was appreciated. The following day, the patient was taken to the operating room for neurosurgical intervention. An entry point just above the transverse sinus in the temporal occipital region was planned. A single bur hole was made, and a needle was inserted along the preplanned trajectory. Evacuation via biopsy syringe revealed frank pus. The procedure was tolerated without complication. No identifiable organism was isolated in the aspirate despite ribosomal sequencing. Dentistry was consulted and evaluated for dental infection and found no evidence of abscess or periodontitis. He was discharged on IV vancomycin and ampicillin-sulbactam for four additional weeks to complete a total antibiotic course of six weeks. Significant improvement was observed on repeat MRI four weeks and then twelve weeks after discharge. He was followed closely as an outpatient during this time and beyond.


A 91-year-old male visited our clinic with right-sided shoulder and neck pain. He was a coach of cricket team by profession. On physical examination, a lump on his right shoulder was found just above the AC joint. He had the history of lump since one year but did not feel any pain, and it continued to enlarge. The lump was nontender, nonmobile, firm, and painless. He felt difficulty in raising his right arm and restricted shoulder movements. Even he was not able to lie on his right side due to pain. There was no history of fever, weight loss, or other symptoms. The medical history was unremarkable, and there was no history of previous surgery on the affected shoulder.
During ultrasound examination, the lump was revealed as cystic lesion just above the AC joint. The size of the cyst was 8.0 × 4.5 × 2.1 cm with posterior acoustic enhancement. The cyst was anechoic with acoustic streaming and has communication with the ST (). Furthermore, the attenuation was seen in the subscapularis tendon. The posterior aspect of the ST was seen hypoechoic with dispersed calcification and posterior acoustic shadowing, suggestive of full-thickness chronic ST tear (). The tear was associated with bony spurs (degenerative changes). A small effusion was seen in the glenohumeral (GH) space, having communication with the subacromial bursa. On real time ultrasound examination, a synovial fluid was observed escaping from the rotator cuff defect across the subacromial bursa and decompressing superiorly through the AC joint. This type of fluid eruption is termed as “geyser sign” by the radiologist. The long head of biceps tendon was present within the bicipital groove and appeared hypoechoic suggesting an element of tendinopathy ().
The subscapularis and infraspinatus tendon demonstrated decreased echogenicity and loss of the usual fibrillar echotexture suggesting tendinopathy (). The infraspinatus tendon also appeared irregular, suggesting detachment of a few fibers (). There was a complete tear (full thickness and full width) of the ST with retraction of 4 cm. Marked distension of the subacromial-subdeltoid bursa was observed (), and the humeral head was also irregular. The swelling over the apex of the shoulder corresponded to a large cystic collection with internal echoes. The fluid was continuous with the AC joint. This presentation is commonly associated with a complete tear of the ST.
MRI was then performed for the confirmation of the ultrasound diagnosis. The mass was visible as hyperintense on T2-weighted scan and hypointense on T1-weighted scan. The fluid was observed in the GH space having communication with the right subacromial bursa. A full-thickness tear in the posterior part of ST was also observed. The lesion appeared as cystic with peripheral enhancement.


A 51-year-old man with relapsed lambda light chain multiple myeloma (MM) was hospitalized due to uremic encephalopathy. Three years prior he experienced remission from MM after treatment with lenalidomide, bortezomib, and dexamethasone (RVD). At that time, he declined maintenance therapy and autologous stem cell transplant. After a relapse involving his cervical spine, he underwent de-compressive surgery, radiation, and restarted RVD. Subsequent bone marrow biopsy revealed disease progression. Dermatology was consulted regarding asymptomatic erythematous papules, plaques, and nodules involving his upper trunk, face, and scalp that had abruptly appeared over the prior three weeks (). Punch biopsies from the chin nodule and a scalp plaque were obtained (). The papillary and reticular dermis were replete with atypical plasma cells, focally resembling plasmablasts, positive for CD138 () and CD45 () by immunohistochemistry. A Grenz zone underlined the epidermis and foci of malignant cells involving adnexal structures including pilosebaceous units were apparent. Dutcher bodies were rare; no Russell bodies were appreciated. In situ hybridization (ISH) confirmed lambda light chain restriction (Figures and ).
The patient's renal failure was attributed to myeloma kidney; and hemodialysis, apheresis, and daratumumab were initiated. While his encephalopathy initially improved, the patient declined clinically and he was transitioned to comfort care three weeks after the biopsy, dying 5 days later.


A 54-year old male patient was admitted to the hospital with a sudden onset of chest pain and backache for 8 h. Physical examination showed blood pressure (BP) of 126/73 mmHg on the upper right arm, 136/69 mmHg on the upper left arm, 158/77 mmHg on the lower right limb, and 146/70 mmHg on the lower left limb. Heart rate was 110 times/min with normal breath sounds in both lungs. No murmur heard upon auscultation. His abdomen was soft and non-tender. There was no edema in both lower limbs. Electrocardiogram (ECG) showed a sinus rhythm of 101 beats/min. Echocardiography (ECHO) showed that the left atrium was 42 × 54 mm, the right atrium was 38 × 48 mm, the right ventricle was 18 mm, the pulmonary artery was 23 mm, and the left ventricle was 53 mm with a left ventricular ejection fraction (LVEF) of 56%. The findings suggested ventricular septal thickening with mild to moderate insufficiency over the left atrial aortic valve. Arterial blood gas analysis showed PaO2 40.8 mmHg, PaCO2 64.0 mmHg, SpO2 92.8%, and Hgb 13.4 g/L. Aortic computed tomography angiography (CTA) confirmed a Stanford type B aortic dissection with an aberrant right subclavian artery (Fig. , , and ) and the aortic diameter was 6 cm.


The patient, a 27-year-old Han Chinese woman, was admitted to our department on July 13, 2017, due to a menstrual disorder of 4 years’ duration. She had experienced a reduction in menstrual frequency 4 years prior, without any obvious cause, with the longest period cycle being up to 4 months and with a decreased amount of menstruation. At the same time, black, coarse hair appeared in her mandible, medioventral line, and areola, and her pubic hair became thicker, accompanied by breast shrinkage. The patient also easily became hungry. No hoarse voice or facial acne appeared. The patient was treated with progesterone at a local hospital, and withdrawal bleeding happened after the treatment. The symptoms recurred after withdrawal of the medicine. No special past history was reported. The patient had menarche at the age of 15, with a period cycle of 28–120 days and a menstrual period duration of 4–5 days. Her most recent menstruation was on June 1, 2017 (after the administration of progesterone). The patient denied a family history of similar diseases. An outpatient test revealed that the patient’s testosterone level was 4.07 nmol/L (reference range, 0.43–2.06 nmol/L). Gynecological color Doppler ultrasound showed bilateral ovaries approximately 44 mm × 23 mm in size on the left side and approximately 50 mm × 26 mm on the right side and more than 10 small follicle echoes inside both ovaries. The diagnosis suggested multiple cystic changes in the bilateral ovaries. Adrenal computed tomography showed that the medial limb of the left adrenal gland was enlarged, with a high possibility of proliferation. The patient’s admission physical examination showed that her body temperature was 36.8 °C, pulse 100 beats/minute, respiration 20 breaths/minute, and blood pressure 119/79 mmHg. The patient’s body mass index (BMI) was 16.89 kg/m2. She had a normal appearance, with no facial acne, full moon face, or buffalo hump and no signs of acanthosis nigricans. Heart, lung, and abdominal examinations showed no abnormalities. Black, coarse hair was observed on the patient’s upper lip, lower jaw, chest, bilateral areolas, and medioventral line of the lower abdomen. The bilateral breasts were symmetrical, and the nipples were slightly invaginated. The pubic hair was thick, and the clitoris was hypertrophied. The Tanner stage was B3P5. The laboratory test results for six gonadal hormones were as follows: luteinizing hormone (LH), 4.96 IU/L (reference range, 1.8–11.78 IU/L); follicle-stimulating hormone (FSH), 2.04 IU/L (3.03–8.08 IU/L); estradiol, 129.0 pmol/L (77.1–921.2 pmol/L); progesterone, 1.03 nmol/L (< 0.318–0.954 nmol/L); prolactin, 494.0 mIU/L (108.8–557.1 mIU/L); and testosterone, 3.40 nmol/L. The patient’s cortisol level was normal, and the secretion rhythm was present and could be inhibited by an overnight 1-mg dexamethasone suppression test. The oral glucose tolerance test (OGTT) result reached the diagnostic criteria for diabetes, and the levels of synchronized insulin were significantly elevated (0 minutes, 701.70 pmol/L; 120 minutes, > 6945 pmol/L). The patient’s dehydroepiandrosterone concentration was 7.44 ng/ml (0.80–10.50 ng/ml); dehydroepiandrosterone sulfate was 27.20 nmol/L (18.00–144.00 nmol/L); 17-hydroxyprogesterone (17-OHP) was 1.44 ng/ml (0.05–1.02 ng/ml); and human anti-Müllerian hormone was 16.40 ng/ml (2.80–6.30 ng/ml). Karyotype analysis showed a karyotype of 46,XX. The patient’s liver and kidney function, blood lipids, and thyroid function were normal. A recheck of the gynecological color Doppler ultrasound data showed a polycystic state of both ovaries and small cysts in the cervix. No abnormality was revealed by urinary system and abdominal color ultrasound. The patient was diagnosed with PCOS with type 2 diabetes. Considering that she was a nonobese patient with PCOS and had an LH/FSH ratio of 2.43 with an abnormal LH pulse frequency, pulsatile gonadotropin-releasing hormone was administered via a pump, which can continuously infuse gonadorelin subcutaneously to restore hypothalamic-pituitary-gonadal axis function and promote menstrual cycle recovery with ovulation. At the same time, metformin was administered to improve insulin sensitivity. After 1 year of follow-up at monthly intervals, the testosterone level of the patient remained above normal and fluctuated between 2.37 and 2.63 nmol/L. Her menstrual disorder, hirsutism, and other symptoms did not improve. Reexamination on July 19, 2018, showed testosterone of 2.74 nmol/L and fasting insulin of 464.50 pmol/L. We considered that the previous diagnosis might have been incorrect, and the patient was hospitalized again on July 31, 2018. A supplementary medical history was collected and showed that the patient’s parents were not close relatives and that the father had type 2 diabetes and was treated with metformin. The mother and three brothers denied a history of diabetes. OGTT was performed on the patient and her mother, and a steamed bread meal test was performed on the father; the results are listed in Table . A human insulin receptor (INSR) antibody was purchased from Wuhan Huamei Bioengineering Co., Ltd. in Hubei Province, and an enzyme-linked immunosorbent assay (using a KHB ST-360 microplate reader) showed negative results for serum INSR antibodies.
After obtaining consent of the patient and her family and a signed informed consent form, 5 ml of peripheral venous blood was collected from the patient and her parents, and genomic DNA was extracted using an ultrasound method. The xGen Exome Research Panel version 1.0 (Integrated DNA Technologies, Coralville, IA, USA) was employed for whole-genome exon capture, and the obtained DNA fragments were then subjected to high-throughput sequencing using the NovaSeq 6000 system (Illumina, San Diego, CA, USA). The raw image files were processed using bcl2fastq (Illumina) to generate raw sequencing data. Low-quality reads, with a quality score less than 20, were filtered out. The resulting sequences were aligned with the human genome reference sequence (hg19), provided by the National Center for Biotechnology Information, using the Burrows-Wheeler Aligner. Single-nucleotide polymorphisms, insertions, and deletions were analyzed in the sequences using SAMtools and Pindel. The data interpretation rules referred to the classification criteria and guidelines for genetic variations of the American College of Medical Genetics and Genomics. The results of the genetic testing showed that the patient and her father had a heterozygous missense mutation, c.3601C>T, p.Arg1201Trp, in the INSR gene (NM_000208) (Fig. ). The gene sequence of the mother was normal at the same locus. Further sequencing of the above locus in the patient’s three brothers revealed that two of them also had the mutation at the same site; however, clinical data of the three brothers have not been obtained yet. The family pedigree is shown in Fig. .
Comprehensive evaluation of the patient’s clinical manifestations, laboratory test results, and gene sequencing led to a clear diagnosis of TAIRS. The treatment regimen was changed to pioglitazone (30 mg once daily) and spironolactone (20 mg twice daily). After 2 months, the patient’s blood potassium level was 4.57 mmol/L, testosterone was 2.91 nmol/L, 0-minute insulin was 299.40 pmol/L, and 120-minute insulin was > 6945 pmol/L. The patient still had no menstruation and no improvement of her hirsutism symptoms. The dosage of spironolactone was adjusted to 40 mg twice daily. On March 20, 2019, the patient’s blood potassium was 4.65 mmol/L, testosterone was 3.08 nmol/L, and fasting insulin was 411.00 pmol/L. The treatment regimen was modified to metformin (0.5 g twice daily), pioglitazone (30 mg once daily), and flutamide tablet (0.0625 g once daily).


A 30-year-old man (60 kg, 159 cm) presented for resection of a glioblastoma multiforme (World Health Organization [WHO] grade IV). There were no pre-existing comorbidities, no allergies, and no history of medication or drugs. The glioblastoma was diagnosed incidentally after a sports accident. The patient had undergone a first uneventful craniotomy 3 weeks earlier for tumor biopsy. There had been no preoperative complaints such as headache, paresiae, dizziness, or seizures. All preoperative laboratory data were normal.
The patient did not receive any preoperative anxiolysis or anti-epileptic drugs, according to our internal standards.
On the day of surgery, we established vascular access and monitored the patient according to American Society of Anesthesiology (ASA) standards in the induction suite. Induction of anesthesia with TCI propofol (Schnider) and remifentanyl (Minto) was smooth, and intubation was uneventful. We placed an arterial line in the left radial artery and positioned 3 electrocardiogram electrodes on the patient's forehead (Fpz, F9, and F10 positions) for DoA monitoring. The electrode impedances were below 6000 Ω.
For intraoperative neurophysiological monitoring and mapping, the ISIS system (Inomed Co., Teningen, Germany) equipped with a constant current stimulator used. Motor evoked potentials (MEP) were elicited via transcranial electrical stimulation and, after dura opening, with a strip electrode placed on the precentral gyrus. Details about our neurophysiological set-up have been published elsewhere.[
Ninety minutes after induction, remifentanil was increased to a target blood concentration of 10 μg/mL to provide stable hemodynamics during Mayfield frame application. Propofol target concentration was adapted to show the characteristic frontal EEG signs of unconsciousness with delta (0.1–4 Hz) oscillations and alpha oscillations (8–12 Hz).[ No local anesthetics were used for incision.
During the dura opening, the patient suddenly suffered tonic-clonic seizures with a motor component, captured by intraoperative EEG (Fig. , arrow 1) and electromyography (EMG) (not shown). We immediately increased the propofol-remifentanil TCI infusion to induction dosage. Once the seizure stopped, we administered levetiracetam 1000 mg i.v. to increase the convulsive threshold. Our EEG monitor displayed an increase in relative beta power shortly before the seizure (Fig. ) and we hypothesized that the plane of anesthesia was too superficial.
Thirty minutes after, the patient developed a second episode of IOS (see Video, Supplemental Video [The video shows the raw EEG signal, captured by the Narcotrend, just before and during the second seizure], showing the raw EEG signal, captured by the Narcotrend, just before and during the second seizure and Fig. , arrow 2). We immediately increased the propofol-remifentanil TCI infusion and administered thiopental until the EEG displayed burst suppression. We then started dexmedetomidine 2 μg/kg/h as a continuous infusion. In retrospect, we could see a pattern of beta power increase shortly before the second seizure in the Narcotrend again (Fig. A).
Ninety minutes after the first seizure, a third episode of IOS clinically manifested under a burst suppression EEG (not shown). A bolus dose of propofol was used to end the seizure, which stopped after 30 seconds. We added phenytoin 750 mg i.v. as anticonvulsant and repeated levetiracetam 1 g i.v. The complete duration of surgery was 7 hours and 45 minutes and no further seizures occurred.
Afterwards, the patient was transferred to an intensive care unit and extubated uneventfully overnight. He showed a left-sided hemiplegia immediately after surgery which improved in the course of the following days. No more seizures were reported. One year after surgery, he is no longer on anti-epileptic drugs, had no further convulsions, and has returned to work. He has only fine motor impairment of the left hand.
The relative spectral power display from our EEG monitor showed a progressive increase in relative power in the beta-wave range, accompanied by a reduction in relative power in the delta-wave range preceding the first 2 seizures (Fig. A). This occurred 8 to 10 minutes before the motor component. To enable more finely grained spectral observations, we also created a spectrogram using Matlab (Matlab R2017b, The Mathworks, Natick, MA) with a moving window of 2 seconds (1 second overlap) (Fig. C). There was a distinct increase in mean absolute beta power immediately prior to the first seizure, but not in the alpha, theta, and delta bands. The EEG immediately prior to the second seizure was characterized by broadband noise. Both seizures were characterized by increased mean absolute delta, theta, and beta power. The slow increase in relative beta power prior to the seizures appears to be associated with a broadening alpha oscillation.
The Bern Cantonal Ethics Committee (Kantonale Ethikkomission [KEK] Bern) waived the need for ethics approval (Req-2018-00985) because the case report does not fall into the Swiss Law for Human Research (Art.2) and the patient gave written informed consent for publication.


Our patient was a relatively healthy 59-year-old African-American male who presented with several weeks of pruritus, jaundice, nausea, and weight loss. His past medical history was significant for hypertension and he did not consume alcohol, IV drugs, or herbal supplements. Physical findings included jaundice, without signs of chronic liver disease, and a benign abdomen without hepatosplenomegaly or ascites. Labs showed elevated liver enzymes in an obstructive pattern ().
He underwent extensive testing for infectious causes which was negative, including hepatitis A, B, C, E, leptospirosis, VDRL, and peripheral smear performed for malaria. Further evaluation including ANA, Smooth muscle antibody, Antimitochondrial antibodies, IgG elevated antibodies, alpha-1 antitrypsin, and carbohydrate antigen 19-9 were all negative. Ultrasound showed only hepatic steatosis, though further abdominal CT showed an enlarged celiac axis lymph node (>1 cm) and portal lymphadenopathy. There was no duct dilatation or either intrahepatic or extrahepatic ducts on endoscopic ultrasound. Cytology from the enlarged celiac node showed small lymphocytes, not diagnostic for lymphoma. An ultrasound-guided liver biopsy was therefore performed ().
This showed noncaseating granulomas and bile duct damage, highly suggestive of sarcoidosis. Sarcoidosis was further confirmed with high activity of angiotensin-converting enzyme and mediastinal/hilar lymphadenopathy in CT of the chest. The patient was started on budesonide and 6-mercaptopurine therapy with marked improvement of symptoms. Repeat labs showed resolution of ACE inhibitor levels and liver chemistries except ALP (). He underwent spirometry which showed a mild reduction in FEV1/FVC (66%). After approximately 8 months of being asymptomatic on treatment, he was seen in the emergency room for complaints of yellowing of skin, itching, pale stools, and weight loss, reportedly going on for the past 6 months. Laboratory findings showed worsening hepatic function with severe direct hyperbilirubinemia. He was started on methylprednisolone for a suspected sarcoidosis flare-up, with no improvement. He underwent an abdominal ultrasound which showed a dilated common bile duct measuring 1.6 cm and biliary sludge, which was not present before. Magnetic Resonance Cholangiopancreatography revealed persistently enlarged perihepatic and periportal lymph nodes and a mass in the ampulla of the pancreas ().
Endoscopic Retrograde Cholangiopancreatography showed a biliary stricture and a double-duct sign (proximal pancreatic and common bile duct dilation) with biliary stricture biopsy displaying a cluster of atypical glandular cells, likely adenocarcinoma (Figures and ). Workup for malignancy included CT PET showing multiple hepatic lesions, the largest measuring 1.3 cm along with other known lymphadenopathy and pancreatic mass. Staging diagnostic laparoscopy showed no evidence of carcinomatosis during which a wedge liver biopsy was taken which showed granulomatous hepatitis, but keratin cocktail immunohistochemistry was negative for carcinoma. He underwent a pancreaticoduodenectomy and antrectomy with no complications.
The pancreatic mass biopsy confirmed pancreatobiliary origin of carcinoma. Three out of 28 lymph nodes were positive for malignant cells and showed evidence of sarcoidosis. The final staging was pT3b pN1, stage 3A. Immunohistochemistry studies revealed a mismatch repair proteins expressed, MSS. He was started on adjuvant chemotherapy and pancreatic enzyme replacement.


A 75-year-old woman presented to a local emergency department with a chief complaint of lip swelling, which had resolved prior to evaluation without any interventions. She had a recent hospitalization a month prior to presentation and was treated for heart failure. Her medication list revealed that she had been discharged on digoxin. Her past medical history was pertinent for heart failure with a reduced injection fraction with a ventricular pacemaker in place.
She presented with mild chest pain. Initial vitals included blood pressure 98/28 mmHg, heart rate 104 beats per minute, respiratory rate 18 breaths per minute, and oxygen saturation of 94% on 3 L/min of oxygen via a nasal cannula. Exam revealed a 2/6 systolic murmur, a pacing device in the chest wall, dry mucous membranes, and disorientation to place and situation, which was reported to be her baseline mental status per her family members.
EKG showed a ventricular paced rhythm at a rate of 96 (Figure ). Lab results included potassium 4.8 mmol/L (normal range 3.5-5.- mmol/L), creatinine 1.2 mg/dL (normal range 0.7-1.3 mg/dL), troponin 0.08 ng/mL (normal <0.03 ng/mL), and digoxin 13.5 ng/mL (therapeutic window 0.8-2.0 ng/mL). After a discussion with the family and patient, the decision was made to treat the patient with supportive care in the emergency department (ED). After initial management, she was admitted and remained asymptomatic during her hospital stay. Her digoxin concentration trended down at the expected rate (Figure ). It was recognized that the patient had mistakenly been taking a 10-fold overdose of digoxin daily since she had filled her prescription (6.25 mg daily vs 0.625 mg daily). She was discharged on hospital Day 6 in good condition.


A 48-year-old male with medical history of major depression disorder, type 2 diabetes mellitus, and diabetic neuropathy presented with exertional dyspnea and cough productive of clear sputum for 4 weeks. He also had pleuritic chest pain, low-grade fever, chills, malaise, and 10 lbs weight loss over the same period of time. He had 12 pack-year smoking history and quit 2 years ago but denied any alcohol or recreational drug use. No history of contact with sick individuals or recent travel. His home medications were amitriptyline, duloxetine, gabapentin, and insulin. He works as a street artist using acrylic airbrush paints for almost 20 years. He was using personal and respiratory protective equipment when handling spray paint materials such as protective goggles, paint masks, and single-use gloves.
On physical examination, he was in mild respiratory distress and tachypneic, with bilateral basilar coarse crackles. His oxygen (O2) saturation was 86% on room air, corrected to 93% through Venti-mask O2 40%. Arterial blood gas showed pH 7.46, pCO2 (partial pressure of carbon dioxide) 28 mm Hg, and pO2 (partial pressure of oxygen) 64 mm Hg. White blood cell count was 11 000 with 5% eosinophils, absolute eosinophils count 550/mL, elevated C-reactive protein, and procalcitonin was negative. On the subsequent day of admission, his white blood cell count trended up to 17 000, with absolute eosinophils count of 1300. Human immunodeficiency virus screening, anti-neutrophil cytoplasmic antibodies—perinuclear pattern, antinuclear antibody, lactate dehydrogenase, mycoplasma antibodies legionella urine antigens, Strongyloides immunoglobulin G, and serum Aspergillus antigen were all negative.
Computed tomography scan of the chest at time of presentation showed bilateral airspace disease, predominantly in lower lobes, with patchy areas of ground-glass densities in a peripheral, peribronchial, and basilar distribution ( and ). He was then started on 1 mg/kg/day of methylprednisolone due to the high suspicion of eosinophilic pneumonia. However, his respiratory distress worsened, and his oxygen requirement continued to increase, that is, he was placed on high-flow oxygen through Vapotherm. Due to the patient’s respiratory distress and high oxygen requirements, bronchoscopy was considered a high-risk procedure; therefore, open lung biopsy through video-assisted thoracotomy was obtained for a definitive diagnosis. Pathological examination of right wedge lung biopsy revealed mixed alveolar inflammatory infiltrate with significant eosinophilic-predominance, multinodular fibroblastic proliferation, and subpleural dense fibrous deposition (), a pattern consistent with CEP and evolving organizing pneumonia.
The diagnostic thoracotomy was complicated by left apical pneumothorax requiring chest tube placement. Despite of the administration of 2 rounds of high-dose steroids, that is, 1 mg/kg/day methylprednisolone for 2 weeks followed by pulse steroid dose of 1 g/day methylprednisolone for 5 more days, the patient continued to have severe respiratory distress requiring non-rebreather mask, high-flow nasal cannula, and, at times, noninvasive positive pressure ventilation. Then, he developed respiratory muscle fatigue and hypoxic hypercapnic respiratory failure requiring endotracheal intubation. He stayed on the ventilator for 2 more weeks and received another round of pulse steroid dose with no significant improvement. A repeat computed tomography chest scan, done 5 weeks later, showed diffuse bilateral interstitial infiltrates, interval worsening of ground-glass opacities scattered throughout both lungs, and evolving fibrotic changes bilaterally ().
Unfortunately, adequate clinical recovery was never achieved during the hospital course and the patient remained in moderate respiratory distress with high oxygen requirements, that is, 4 L O2 via nasal cannula or Venti-mask 35% interchangeably. The patient then opted for a palliative level of care and was discharged to a hospice unit where he passed away 3 weeks after discharge.


This 53-year-old male presented to our office with complaints of an enlarging right knee mass causing intermittent, vague pain for the past couple months. He denied trauma to right knee, numbness, tingling and weakness. He reported vague knee pain with knee flexion and to palpation. Patient had a 2 × 2 cm firm, tender, mobile mass on right anterior knee. A neurological examination revealed intact motor and sensation. No muscle atrophy or knee effusion is present on physical examination.
Patient received general anesthesia prior to incision. A 3-cm skin incision was made on the mass, which was deepened down through soft tissues. The patellar capsule was opened and an oblong mass identified. The mass was dissected free from the patellar capsule and suture ligated at the base of the stalk.
Macroscopically, the tumor appeared encapsulated, rubbery and pink-tan in color. Microscopic examination reveals that tumor composed of biphasic spindle hypercellular Antoni A areas and hypocellular Antoni B areas (Fig. ). Higher magnification reveals that spindle tumor cells are narrow, elongate, wavy with tapered ends interspersed with collagen fibers (Fig. ). Immunohistochemical staining revealed that most tumor cells reacted strongly for S-100 protein (Fig. ). Combined with immunohistochemical profile, these histological features are diagnostic of benign schwannoma. Patient was discharged home on the same day as the operation. At the 1-month follow-up, he was symptom free, without any pain or sensory disturbances.


A proband, female, aged 10 years and 3 months, with a medical history of recurrent fever and abdominal distention prior to the age of 5 years and 9 months, was admitted to our hospital with recurrent fever and cough for 3 days. Five years and nine months before admission to our hospital (when the child patient was 4 years and 6 months old), she was diagnosed with Epstein–Barr virus (EBV)–HLH due to fever, hepatosplenomegaly, pancytopenia, increase of ferritins and positive EBV-DNA, and thus was treated based on the HLH-2004 plan for 8 weeks. Treatment was then stopped, and the disease remained in continuous remission for 22 months. Three years and nine months before admission to our hospital, the patient was diagnosed as having a relapse of EBV–HLH due to the aforesaid symptoms and her positive blood EBV–DNA, and she was treated again by chemotherapy based on the HLH-2004 plan for 8 weeks. Symptoms relieved and she stopped taking drugs, and the duration of the disease remission reached about 19 months. Two years before admission to our hospital, the child patient developed recurrent fever and abdominal distension, which improved after receiving anti-infective therapy and taking dexamethasone (details not known) in a local clinic. She denied having a history of infectious disease and has been inoculated with hepatitis B vaccine, BCG vaccine, poliomyelitis attenuated live vaccine, diphtheria pertussis tetanus vaccine, measles attenuated live vaccine, and epidemic encephalitis B vaccine, without any abnormal reaction. The patient was born in full term, G3P3, her parents’ marriage was consanguineous (Fig. ), and the two elder brothers respectively developed recurrent fever and abdominal distension symptoms similar to the patient's at the ages of 4 and 5 years and died of treatment failure in another hospital. Specific diagnosis and treatment as well as causes of death were unknown. After admission to our hospital, the child patient had a continuous high fever (39–40 °C), with an acute serious sickly appearance, without stained yellow skin or rash, but scattered ecchymosis was found. The superficial lymph nodes were not affected but her tonsils were slightly swollen. Medium and fine moist rales were heard in her right lung. Her abdomen was distended, the liver was 3.0 and 5.0 cm below the costal margin and the xiphoid, respectively. No abnormity was found in her nervous system. The hemogram revealed pancytopenia with the neutrophils 0.29 × 109/L, Hb 52 g/L, and platelets 6 × 109/L, respectively. The fibrinogen was decreased (the lowest value was 44 mg/dL), and the ferritin was elevated (995.3 ng/mL). Her triglyceride was normal, and a hemophagocytic phenomenon was found in a bone marrow examination. Multiple etiological examinations (blood culture, fungus G test and GM test, mycobacterium tuberculosis T-SPOT, mycoplasma, chlamydia and EBV-DNA [six repeated examinations], cytomegalovirus [CMV]-DNA, TORCH and seven combined examinations of respiratory viruses) were negative. According to eight HLH clinical diagnosis standards formulated by the International Histiocyte Society in 2004, the patient was clinically diagnosed as HLH. Due to a positive family history, in order to further specify the causes of the disease, we examined the activity of NK cells, the CD107a degranulation function of cytotoxic T lymphocytes and NK cells, as well as hereditary HLH-related gene mutations, and found that:
the activity of NK cells in the proband was reduced slightly, the CD107a degranulation function in cytotoxic T lymphocytes and NK cells also was obviously declined, and her parents also presented with reduced NK cell activity and slightly declined CD107a degranulation in CTL and NK cells (Fig. );
a homozygous mutation in STX11 (c.49C>T), which converted the codon encoding the glutamine amino acid at position 17 in syntaxin11 to a termination codon (p.Q17X), leading to early termination of the synthesis of syntaxin11 and loss of 240 amino acids (Fig. ).
This mutation was inherited from the parents of the proband (Figs. and ), and the result proved that the proband suffered from FHL-4. After admission to our hospital, the patient found relief after receiving treatment based on the HLH-2004 protocol; she has been alive for 6 years and 10 months since the onset, has stopped taking the dexamethasone and etoposide, but still takes cyclosporine orally to maintain treatment. She has undergone HLA matching and now is actively looking for a donor to undergo a hematopoietic stem cell transplantation.


This 53-year-old retired man presented with bilateral thigh, buttocks and back pain for 2 months prior to admission. The pain was intermittent, which became worse at night seriously affecting his sleep. He also felt back weakness, could not keep his back upright while walking. He had a history of splenectomy 11 years ago, due to moderate anemia caused by an unexplained splenomegaly. He had no bone pain or neurological damage at that time. Postoperative pathological results showed only splenic hemolytic enlargement accompanied by iron-containing nodule formation and focal infarction, with no lymphoma or other tumor-like changes. We reviewed the pathological results of spleen and no epithelioid hemangioma like changes was found.
After relevant examination, 99mTc-MDP bone scintigraphy showed multiple metabolism enhancements of the multiple bones; multiple bone metastases were suspected (Fig. ). 18F-FDG-PET/CT showed multiple hypermetabolic skeletal lesions, bone metastasis or metabolic bone disease was suspected (Fig. ). X-ray showed that bone density of cervical vertebrae, thoracic vertebrae, lumbar vertebrae, skull, pelvis, bilateral scapula, bilateral clavicle, bilateral ribs, bilateral humerus and upper femur were generally increased, with multiple high-density and low-density nodules; metabolic bone disease was suspected (Fig. ). Thereafter we performed CT-guided biopsy of the left ilium and right femoral trochanter area, pathological findings indicated that puncture tissue had irregular hyperplasia of thick and thin walled blood vessels; multiple epithelioid hemangioma was considered (Fig. ). The patient had elevated serum bone specific alkaline phosphatase (BALP) and type I collagen cross-linked C-terminal telopeptide (CTX) levels. He was treated with 2 times of intravenous Zometa (zoledronate; Novartis Pharmaceuticals, Basel, Switzerland, 4 mg) with three-month interval, 4 mg each time, which lowered BALP and CTX levels significantly and provided control of the patient's bone pain (Table ). However, the effects were short lived with the pain returning 1 month after the second course of intravenous Zometa; the patient suffered paraplegia and was admitted under the department of orthopedics. Three-dimensional CT suggest multiple bone destruction of the spine and its attachments, T11 compression fracture, with diagnosis of thoracic 11–12 tumor with paraplegia (Frankel A). Post-lumbar thoracic 11–12 tumor resection, spinal canal decompression, bilateral spinal nerve release, bone graft fusion internal fixation was performed (Fig. ). The postoperative pathological results still suggested the diagnosis of epithelioid hemangioma of bone. His pain was controlled for several months. Unfortunately, three months after the surgery, the bone pain aggravated again, which was more obvious at night, with difficult of walking upright, he can only stand with the support of his hands could not stand upright and his activity time was shortened. The patient was given another 2 times of Zometa with three-month interval, with significant improvement of bone metabolic markers (Table ). He also underwent T11 vertebroplasty and the bone pain was significantly relieved. The patient was followed up every 2–3 months to check for any relapse, he was doing well with no significant pain after one year follow up.
This case report was approved by the Ethics Committee of the West China Hospital of Sichuan University, Chengdu, China, and the patient has provided informed consent for publication of the case.


A 31-year-old male was admitted to the hospital with a 4-day history of fever, diaphoresis, and right submandibular edema. He also complained of sore throat, dyspnea, and pleuritic chest pain. He had a past history of HIV infection since 2017 and genital chlamydia infection. He was not taking antiretrovirals due to lack of compliance with his regular follow-up visits and medications. He endorsed alcohol, tobacco, and marijuana use but denied other illicit or intravenous drug use. On physical examination, he appeared acutely ill. He was febrile (39.4°C), tachycardic (143 heart rate), normotensive (121/66 mm Hg), and tachypneic (28 respiratory rate). Oxygen saturation was 99% on room air. He had right submandibular edema and a small orolabial ulcer with mild purulent drainage (). Bilateral tonsillar hypertrophy without exudates was also noted. There were no signs of dental or periodontal infection. Pulmonary examination revealed decreased breath sounds in the left lower lung field and bibasilar crackles. The rest of his examination was unremarkable. Laboratory studies revealed a white blood cell count of 26 200 cells/µL (85% neutrophils, 8% bands). Arterial blood gases and routine chemistries were normal. CD4 count was 356 cells/µL. Chest X-ray showed bilateral nodular opacities and a small left pleural effusion. Computed tomography (CT) scan of the neck showed right-sided submandibular inflammation, facial vein thrombosis, and reactive cervical lymphadenopathy (). CT scan of the chest revealed numerous subpleural and intraparenchymal pulmonary nodules, some with spiculated cavitation (). Small bilateral pleural effusions, a large consolidative opacification in the left lower lobe, and bibasilar ground glass opacities were also noted. CT angiogram showed partial filling defects suggestive of right pulmonary artery and left anterior segment pulmonary embolism.
The patient was started on intravenous vancomycin and piperacillin/tazobactam. Piperacillin/tazobactam was discontinued after isolation of MRSA from blood cultures with no gram-negative or anaerobic growth. MRSA was also isolated from a drainage culture of the superficial orofacial ulcer. A transthoracic echocardiogram showed no valvular lesions or signs suggestive of infectious endocarditis. He initially received anticoagulation with intravenous heparin for 5 days and then oral apixaban for 5 additional days, but it was later discontinued.
The patient responded well to treatment, and after 1 week, his submandibular edema subsided (). He was discharged from the hospital after receiving 2 weeks of intravenous vancomycin and completed a 4-week course as an outpatient. This was administered via a peripherally inserted central catheter at a skilled nursing facility with monitoring of routine laboratories and vancomycin levels. At a 2-month follow-up outpatient appointment, he was doing well. He was instructed to keep regular follow-up visits for his HIV care.


A 26-year-old female, with a history of primary infertility, underwent follicular stimulating hormone therapy. On follow-up, she had a positive serum beta-human chorionic gonadotropin (hCG) test. On transvaginal ultrasound, an intrauterine gestational sac of 3 mm was noted (corresponding to the gestational age of approximately four weeks). After four weeks, the patient presented with right lower quadrant abdominal pain. She was hemodynamically stable. On transvaginal ultrasound, a right adnexal complex lesion (echogenic ring sign) was noted, in addition to the intrauterine gestational sac. On the Doppler study, the ring of fire sign was noted, suggesting ruptured ectopic pregnancy (Figure ).
Thus, the diagnosis of heterotopic pregnancy was established. The patient underwent laparoscopic right total salpingectomy. Histopathology examination confirmed the diagnosis of an ectopic pregnancy. The patient underwent repeat ultrasound after one week that showed no fetal pole and confirmed missed abortion of intrauterine pregnancy (IUP).


A 37-year-old female (G3P0A2) with a history of an ectopic pregnancy managed conservatively presented in the emergency room with lower abdominal pain. On examination, the patient's vitals were stable, and there was tenderness in the left lower abdomen. Transvaginal ultrasound showed a non-viable IUP and a viable left adnexal ectopic pregnancy (Figure ).
These findings confirmed the diagnosis of heterotopic pregnancy. A left salpingectomy was done via a mini-laparotomy for the ectopic conceptus, and suction evacuation of the uterus was done as well.


A 43-year-old female (G8P4A3) presented to the emergency department with mild vaginal bleeding and lower abdominal pain. On examination, the patient was alert and vitally stable. High-resolution transvaginal ultrasound (US) confirmed an incomplete abortion. On color Doppler, the ring of fire sign was noted in the left adnexa (Figure ).
These findings suggested the diagnosis of heterotopic pregnancy. The patient underwent suction and evacuation with laparoscopic left salpingectomy on the same admission. On follow-up labs, the beta-hCG showed a significant downward trend.


A 40-year-old female (G4P2A1) with a history of secondary infertility underwent ovulation induction treatment (follicle-stimulating hormone injections). She presented in the emergency department with lower abdominal pain. On examination, her vitals were stable with mild abdominal tenderness. Her beta-hCG was positive. Transvaginal ultrasound showed a non-viable intrauterine pregnancy and right adnexal ectopic pregnancy (Figure ).
These findings were suggestive of heterotopic pregnancy. Laparoscopic right salpingectomy was performed, and follow-up beta-hCG showed a downward trend.


A 30-year-old female (G2P1A0) with no significant past medical history presented to the emergency department with abdominal pain. On examination, her vitals were stable. Her beta-hCG was positive. Transvaginal ultrasound showed a viable intrauterine pregnancy and an ectopic pregnancy in the right adnexa with a possible surrounding hematoma (Figure ).
A laparoscopic right salpingectomy was done for this patient. Regular obstetric follow-up was done for the intrauterine pregnancy, and at term, the baby was delivered via spontaneous vaginal delivery with no significant complications.


A 30-year-old female (G2P0A1) with a history of in vitro fertilization (three embryos transferred) presented with lower abdominal pain, vomiting, and vaginal discharge. On examination, she was hemodynamically stable. Transvaginal ultrasound showed a viable intrauterine pregnancy and a left adnexal ectopic pregnancy with surrounding hematoma suggestive heterotopic pregnancy (Figure ).
A laparoscopic left salpingectomy was done, and the intraoperative findings confirmed ruptured ectopic. On follow-up ultrasound, an incomplete abortion of the IUP was noted.


A 65-year-old man with a 60-pack-year smoking history presented with a right middle lobe ground glass opaque (GGO) nodule which measured 1.9 cm. One year later, the nodule progressed in size to 2.8 cm (Figure ). CT scan showed there was no mediastinal adenopathy present in the chest. Transthoracic needle aspiration was performed and demonstrated atypical adenomatous hyperplasia (AAH) (Figure ). The patient was recommended to undergo right middle lobectomy, but refused and was referred for a radiation oncology consultation. The case was presented at the multidisciplinary tumor conference where there were multiple differing opinions on the future management considerations for this patient. To date, there is no evidence supporting stereotactic body radiotherapy (SBRT) use for AAH. Observation was recommended with a serial CT scan every three months. At the two-year follow-up visit with repeat chest CT, the GGO is unchanged in appearance or size.
In another case, a 73-year-old female with a long smoking history was referred to thoracic surgery for a slow-growing nodule in her left upper lobe. A CT scan showed a 2.3 x 1.3 x 0.9 cm GGO nodule in the anterior left upper lobe (Figure ). At this time, a three-month follow-up was recommended. A repeat scan showed unchanged size and configuration of the nodule. The patient was recommended to follow-up in six months, but she did not return for almost a year. At that time, a biopsy was performed showing alveolar atypia consistent with AAH (Figure ). A repeat CT was performed six months following the biopsy and the nodule became "semisolid" with both solid and GGO features here (Figure ). At no time did any of the CT scans demonstrate mediastinal adenopathy or other abnormality. The patient's case was presented at the multi-disciplinary tumor board and surgical consultation was recommended.
Upon presentation to thoracic surgery, the patient continued to smoke. She denied any symptoms of cough, weight loss, or change in appetite. Given the location and concerns for potential malignancy within the previously identified atypical adenomatous hyperplasia, the patient was consented for lobectomy. The patient underwent an uneventful left upper lobectomy and lymph node dissection. Final pathology showed two foci of invasive moderately differentiated adenocarcinoma, acinar pattern (Figure -). Multiple lymph nodes were positive for malignancy including 4L, 11L and other peribronchial nodes with no evidence of distant metastases or brain metastases on CT and PET scan. The patient’s final pathology demonstrated her to be T3N2M0. She was referred to medical oncology for chemotherapy and radiation oncology for radiation.
Together these cases demonstrate the varying opinions on clinical management of AAH and to date, there is no empiric treatment for AAH. Traditional treatment options include surgical resection or observation with serial CT scans. The role of SBRT is unknown due to lack of data on the use of SBRT in treating AAH. In addition, the latter case demonstrates that AAH can act as precursor lesion of adenocarcinoma.


A 75-year-old Caucasian female presented to the emergency room (ER) in January for a change in mental status and confusion after she was found wandering outside her home. The patient complained of generalized myalgias, nonproductive cough, and runny nose in the preceding few days for which she called her primary care physician and was given a script of oseltamivir, attributing the symptoms to influenza virus infection. Pertinent past medical history included hypertension, chronic kidney disease (CKD) stage III, with the baseline estimated glomerular filtration rate (eGFR) 45 milliequivalent/liter, and type II diabetes (DMT-2). Her medications included metformin, pioglitazone, amlodipine, atorvastatin, and ezetimibe. She used to live by herself and didn’t drink or smoke. Vitals in the ER were temperature: 93 F, pulse: 55/min, blood pressure: 96/54 mmHg, oxygen saturation: 98% on ambient air, and respiratory rate: 28/min.
Physical examination showed that she was lethargic and oriented only to self, with dry mucosal membranes and cold, clammy skin. The neck was supple; extraocular movements were intact. Lungs were clear to auscultation. The rest of the examination, including the cardiovascular and gastrointestinal systems, were unremarkable. Pertinent laboratory evaluation, including complete metabolic profile (CMP) showed serum glucose of 187 mg/dL, creatinine: 11.5 mg/dL (baseline 1.8 mg/dl), sodium: 131 meq/L, potassium: 7.9 meq/L, bicarbonate: 5 meq/L, anion gap: 35, and glycosylated hemoglobin (HbA1c) of 6.2 mg/dL. Complete blood count (CBC) showed hemoglobin of 10 g/dL, platelets 370,000/uL, and white blood cells (WBC): 9.3 k/uL. The coagulation profile was normal. The lactic acid level was eight (8) meq/L. Venous blood gas analysis showed pH: 7.009, pCO2: 18.2 mmHg, and bicarbonate level: 5.1 mmol/L. Serum osmolarity was 312 mOsm/kg, with an osmolar anion gap of 12 mOsm/kg. Urinalysis showed glucose of 500 mg/dL, proteinuria of 30 mg/dL. Electrocardiogram (EKG) showed a first-degree heart block and broad QRS complex, as shown in Figure .
Computerized tomographic scan (CT) head and chest X-ray were unremarkable.
She was aggressively resuscitated with intravenous (IV) fluids. Hyperkalemia was treated with IV insulin, dextrose, calcium gluconate, sodium bicarbonate, and inhaled albuterol. Urgent hemodialysis was also arranged. Attributing her acute severe metabolic acidosis to influenza complicated by bacterial superinfection, she was started on broad-spectrum antibiotics and oseltamivir, and workup for acute renal failure, including antinuclear antibody (ANA), antineutrophilic cytoplasmic antibody (ANCA), complement levels, urine eosinophils, and renal ultrasound, was ordered. After hemodialysis (HD), electrolytes improved to a bicarbonate level of 18 mmol/L, potassium of 3.8 meq/L, and a pH of 7.433. She remained hemodynamically stable at this point. Repeat metabolic profile (BMP) fours hour after the completion of HD showed the bicarbonate level dropping down to 13 meq/L again. Endocrinology was consulted due to persistent metabolic acidosis. At this point, the patient recalled that she was also taking dapagliflozin (an SGLT-2 inhibitor) up to two weeks before the presentation when she stopped taking it due to her improving HbA1c.
Her serum beta-hydroxybutyrate result came back at 92.5 mg/dl. She was started on IV insulin and dextrose. Serum beta-hydroxybutyrate started improving and resolved entirely within 15 hours of beginning IV insulin, and bicarbonate improved to 22 meq/l. Later on, her urine output improved and electrolytes stabilized without further need for HD. Creatinine trended down and stabilized at 2.5 meq/L.
She resumed a regular diet, and beta-hydroxybutyrate remained within the normal range. Her blood sugar levels remained in the range of 110-150 mg/dl without hypoglycemic agents. Workup for acute renal failure came back negative. She was discharged to a rehabilitation center with the plan to establish her diabetic regimen, depending on blood sugar levels on an outpatient basis.


Following a 7-day scheduled absence from the workplace, a 34-year old male aluminum smelter pot room process control operator (PCO) returned to work to commence a series of evening shifts from 6.00 PM to 6.00 AM. The first half of the shift involved vigorous and intense pot room control tasks. Following the lunch break (ie, during the second half of the shift), the worker was assigned to other routine, though less demanding, tasks with less heat stress potential.
Within 6 hours of starting his first shift, while performing routine tasks that involved the occasional presence on the “catwalk” between sequential pots (Figure ), he began to experience hand-cramping. Shift safety personnel were notified, and the worker was encouraged to access available fluids. The worker continued to perform his assigned job tasks, however, and near the end of his shift at approximately 6.00 AM, he experienced hand-cramping again, mentioning this to a coworker but not the crew leader or supervisor. He finished his shift and clocked out.
Forty-five minutes later the site plant protection office received a request to send an ambulance to the shower house for a worker with difficulty breathing. Upon arriving at the scene, the dispatched emergency medical technician (EMT) found the operator walking out of the building and describing body cramps but without concern regarding his breathing. He was noted to be diaphoretic. The worker entered the ambulance under his own power, a presumptive assessment of heat exhaustion was made, and first aid attention consisting of oral fluids and rehydration salts was provided in accordance with site protocol. Initial vital signs showed an oral temperature of only 36.5°C (97.7°F), blood pressure of 160/80 mm Hg, pulse rate of 91 beats per minute, respiratory rate of 18 breaths per minute, and pulse oximetry of 98% (SpO2—peripheral oxygen saturation). Recognizing the temperature as unexpectedly low, which was thought secondary to the ingestion of cool rehydration fluids, the EMT intended to repeat this measurement within a few minutes; however, ensuing acute medical events took precedence and a repeat temperature was never obtained in the field.
During transport to the site medical department, the worker experienced a generalized seizure with, as described by the EMT, decorticate posturing, following which he became agitated, combative, and pugnacious. Random blood glucose was 142 mg/dL (7.9 mmol/L). The community ambulance service was summoned, the worker was stabilized with haloperidol and midazolam, and transferred to the proximate regional hospital emergency department for further evaluation and care.
In the emergency department his initial measured oral temperature was 38.1°C (100.6°F), blood pressure 178/89 mm Hg, pulse rate 100 beats per minute, respiratory rate 16 breaths per minute, SpO2 95%, and body mass index (BMI) 34.62 kg/m2. Presenting serum sodium level was 114 mmol/L (reference range, 136-145), with creatinine 1.6 mg/dL (reference range, 0.6-1.2) and white blood cell count 24.0 thousand/uL (reference range, 4.1-10.9). Anion gap was calculated at 31 mmol/L (reference range, 7-14) with serum lactic acid level of 12.4 mmol/L (reference range, 0.5-2.0). The initial total creatinine phosphokinase (CPK) level was 7641 units/L (reference range, 36-174). Microscopic urinalysis indicated the presence of a large amount of blood but was otherwise negative. A urinary myoglobin level was not obtained. Acetaminophen and salicylate levels were within normal limits. A seven-panel urine drug screen was negative for any substances, including amphetamines/methamphetamines or derivatives. Serum alcohol was less than 10 mg/dL (reference range, <10). Arterial blood gas was consistent with metabolic acidosis. The remaining laboratory values upon admission are noted in Table . Admitting diagnoses included: hyponatremia (ICD-10-CM, E87.1), agitation (R45.1), acute delirium (R41.0), acute respiratory failure (J96.00), acute encephalopathy (G93.40), rhabdomyolysis (M62.82), seizure (G40.89), aspiration (J95.89), and lactic acidosis (E87.2).
Airway protection via intubation was achieved. However, he aspirated during the difficult procedure and remained intubated for the first 48 hours of hospitalization. Day-of-admission computerized tomography scan of the head without contrast showed no intracranial mass, hemorrhage, hydrocephalus, or acute intracranial abnormality. An electrocardiogram (EKG) showed sinus tachycardia.
Past medical history was notable for essential hypertension, gastroesophageal reflux disease, and mild attention deficit hyperactivity disorder (ADHD), for which the worker had been prescribed losartan, lansoprazole, and amphetamine/dextroamphetamine, respectively. He was a former smoker and admitted to moderate alcohol ingestion several times per week. He had no known history of prior HRI. However, in the aftermath of this incident, he discovered through family medical history inquiry that he had a history of “cramping” in relation to sports activities and exertion in his youth. There was no known history of hyponatremia or alcohol-use disorders.,
Stabilization treatment included rehydration with normal saline solution per recommendation of the consulting nephrologist. An electroencephalogram (EEG) on admission day 2 was indicative of moderate encephalopathy of nonspecific etiology, but without evidence for epileptiform activity. Enterococcus faecalis sepsis was diagnosed by blood culture on admission day 4. Hepatitis serologies were nonreactive, as were human immunodeficiency virus 1 and 2.
The worker spent a total of 13 days in the hospital, the first five being in the intensive care unit. Total CPK, aspartate aminotransferase (AST), and alanine aminotransferase (ALT) values in units/L peaked at 125 000, 1478, and 552, respectively, on the fourth hospital day, with a progressive decline in values thereafter (Table ).
The hospital discharge summary listed “severe hyponatremia likely triggered by heat exposure” among the discharge diagnoses, with a further notation that the “hyponatremia was likely related to heat stroke.” At the time of discharge, he had pain and weakness in both lower extremities, requiring the use of ankle-foot orthoses to support standing and walking, as well as numbness and tingling of his upper extremities, primarily his hands.
The worker subsequently underwent a diagnostic evaluation for his apparent polyneuropathy. A noncontrast magnetic resonance imaging (MRI) of the head performed 6 weeks after the incident and 4 weeks postdischarge from the hospital revealed an approximately 1 × 2 cm lobulated nonenhancing cerebellar lesion involving the superior aspect of the left half of the vermis. Repeat noncontrast MRI of the head performed at 20 weeks after the incident revealed stable findings, with no change of this cerebellar lesion.
At 32 weeks after the incident, he remained out of work pending further recovery in his musculoskeletal functional status, resolution or stabilization of the neuropathies, and disposition review regarding return to work in a high-heat stress environment.


In June 2017, a 69-year-old male patient, with complaints of fever and cough for over 2 months, was admitted to the First Affiliated Hospital of Wenzhou Medical University, Wenzhou, China. The patient had undergone brain stem tumor resection 1 year ago. He developed symptoms such as fever and cough for 2 months, during which septic shock occurred, causing unconsciousness. Then, he was admitted to the ICU, and treated with cefoperazone-sulbactam, dispersibleco-trimoxazole, meropenem, tigecycline and fosfomycin piperacillin, and tazobactam. Invasive treatment was also performed, such as endotracheal intubation and enteral feeding tube. During hospitalization, 11 K. pneumoniae strains were isolated from multiple specimens (sputum, wound, stool, urine, and blood). Among them, FK4111, FK4623, and FK4758 were separated from stool, and the ST type determined were ST37, ST37, and ST11, respectively. It was worth noting that in January 2018, 10 days after the date of isolation of ST11 Klebsiella pneumoniae from the patient’s stool, the patient developed a bloodstream infection. In June and November 2017, ST37 K. pneumoniae also colonized the intestine but did not cause infection. Death occurred after the patient had bloodstream infection ().


A 57-year-old African American female with a history of alcohol abuse and diabetes
mellitus presented to the emergency department with a 2-day history of severe
diffuse abdominal pain with radiation to the back. Associated symptoms included
nausea and vomiting. She consumed 5 bottles of beer daily with the most recent
alcohol intake 2 days prior to presentation. Vital signs on presentation were blood
pressure 123/90 mm Hg, pulse 125 beats/minute, respiratory rate 17 breaths/minute,
and temperature 36.8°C. Physical examination was significant for a mildly tender
abdomen. Laboratory findings revealed leukocytosis of 14 600/mm3 and
lipase of 882 U/L (normal = 16-62 U/L). Computed tomographic imaging of the abdomen
with and without contrast revealed peripancreatic fat stranding suggestive of acute
interstitial pancreatitis. The patient was admitted for intravenous fluid
resuscitation and pain management.
On day 2 of admission, the patient became dyspneic and hypoxemic (digital pulse
oximetry 82%) on room air. Chest radiography showed pulmonary edema, and abdominal
ultrasound revealed a dilated inferior vena cava. Troponin I 0.97 ng/mL (normal
<0.03 ng/mL) and brain natriuretic peptide 1627 pg/mL (normal <100 pg/mL)
levels were elevated. A 12-lead electrocardiogram (ECG; ) obtained revealed diffuse ischemic
T-wave inversion. The patient was aggressively diuresed with significant improvement
in respiratory status. Transthoracic echocardiography revealed a left ventricular
ejection fraction of 40% with basal segment hyperkinesis but apical akinesis
consistent with stress-induced cardiomyopathy (). Coronary angiography revealed
normal coronary vessels.
The patient’s abdominal pain resolved, and there were no further complications from
her pancreatitis. She was started on an angiotensin converting enzyme (ACE)
inhibitor and a β-blocker and discharged to home in stable condition. A repeat
transthoracic echocardiography obtained 4 months later revealed unchanged left
ventricular ejection fraction and persistent apical akinesis in the setting of
persistent alcohol use. The ACE inhibitor and β-blocker were continued, and she was
counselled on alcohol cessation.


A 34-year-old African (Zimbabwean) woman in her first pregnancy presented to our institution at 22 weeks of gestation by referral from a local clinic with a diagnosis of OI. She was wheelchair-bound. Her medical history revealed several fractures as a neonate and in childhood, leading to a diagnosis of OI based on clinical presentation and examination. Confirmatory tests were not available in the public health sector. She has been in and out of the hospital since childhood because of these fractures, with various treatments having been received, including plaster of Paris casts and traction. She had a fracture of the right femur at the age of 12 years, which made her wheelchair-dependent. She can, however, stand with support and climb onto and off the wheelchair without assistance. She was abnormally short compared with her siblings and peers. She had normal pubertal development, and her menstrual cycle was very regular. Her sexual debut was at 28 years, and she has had one sexual partner. Her boyfriend, who was married and had five other children, contributed little to her welfare. She was a vendor receiving some financial support from her siblings. She took combined oral contraceptives for 4 years; however, she had stopped taking them because she thought she could not get pregnant. The result of cervical cancer screening with visual inspection with acetic acid and cervicography done in 2016 was negative. She had no family history suggestive of OI. She is educated to form 4 (ordinary level). This current pregnancy, though unplanned, was booked at 18 weeks of gestation. The results of antenatal screening for human immunodeficiency virus and syphilis were negative.
On examination, she was of short stature, 120 cm in height, with a triangular face. Her sclera was white. She had poor dentition with grayish discoloration. She had short limbs, with the right lower limb shorter than the left, and deformities were noted on both thighs. She did not have scoliosis or kyphosis. Her chest was barrel-shaped with good air entry bilaterally. She had normal blood pressure and a normal cardiovascular system examination finding. Her abdominal examination revealed central obesity and a bulky uterus of 20-week-size gestation.
A multidisciplinary team was involved in her management. The team included obstetricians, a maternal-fetal medicine specialist, pediatricians, anesthetists, a psychologist, and midwives. Ultrasound performed by a maternal-fetal medicine specialist showed a fetus with a bowed femur and short humerus. No fractures were noted. The fetal skull was easily deformable. No other malformations were noted. The conclusion was that the fetus had OI, nonsevere disease. Pediatricians counseled the patient about the fetal condition and the possibility of preterm delivery and its accompanying complications. With the limitations of the neonatal unit, the conclusion was an unfavorable prognosis for the neonatal outcome. The patient, however, was willing to continue with the pregnancy.
She was subsequently followed closely in the antenatal clinic, with a plan for pregnancy to continue to 37 weeks, with the possibility of early delivery if maternal respiratory compromise occurred. She had an uneventful antenatal period, with serial ultrasound showing satisfactory growth. She had an admission for a urinary tract infection at 29 weeks of gestation and was treated with oral antibiotics with complete resolution. Contraceptive counseling was provided, and she opted for tubal ligation, which was done at the time of cesarean section.
She ultimately delivered, by an elective transverse lower segment cesarean section at 37 weeks, a live female fetus with a birth weight of 2100 g. The operation was done with the patient under general anesthesia, and a smooth intubation was done with care so as not to cause cervical spinal fracture. Bilateral tubal ligation was performed. Estimated blood loss at the time of delivery was 500 ml, and 10 IU of oxytocin was administered for delivery of the placenta. The patient was extubated after surgery and admitted to the high-dependency unit and monitored for 24 hours. The baby was admitted to the neonatal unit. She developed a chest infection, which was successfully treated with intravenous antibiotics. The mother was managed for a paralytic ileus on day 2 after surgery. She was discharged on day 7. She developed a superficial surgical site infection, which was managed with daily dressings and oral antibiotics on an outpatient basis. The child is currently seen in follow-up by her pediatrician for OI. She has blue sclera. She was noted to have healed fractures on the left radius and right femur at 6 weeks of age. Initial neonatal x-rays had not revealed any fractures. Bone deformities were also noted. She can now walk with support. The rest of her developmental milestones are normal. No other fractures have been reported since they were originally noted. The parents are not able to afford genetic tests.


A 26-year-old female presented, walking, to the emergency department with a 2 week history of abdominal pain radiating to the back. This was associated with a 2 day history of nausea and vomiting following an alcohol binge. On one episode the vomitus was noted to be blood stained. She had no prior medical, surgical or family history. She consumed alcohol regularly and had a binge on the two days prior. She consumed Non-Steroidal Anti-Inflammatory Drugs (NSAID) chronically and had an eight-pack-year history of smoking.
On arrival she had a blood pressure of 124/64 mmHg and was tachycardic with a heart rate of 118bpm. She had a temperature of 37 °C. Her urine dipstick and pregnancy tests were negative. On examination she had epigastric tenderness but her abdomen was soft and she had no signs of peritonitis. Her cardiovascular, respiratory and central nervous system examinations were unremarkable. She had no malaena or blood on rectal examination.


Our patient was a 13-yr-old girl who was referred to our hospital because of acute abdominal pain. She had a fever for two weeks before being referred to us. Her menarche had begun one year before her referral. She had suffered from severe dysmenorrhea until then, and her last menstrual cycle had been two days before coming to the hospital. Her abdominal pain had increased during her last menstruation. The patient had a fever, chills, abdominal pain, and nausea at the time of admission to the hospital. She did not have a vaginal discharge with bad odor and had no particular problems in her medical history. Generalized abdominal tenderness was diagnosed in the physical examination, but mild vaginal bleeding and right lateral vaginal wall bulging were found in her vaginal and transrectal examinations. She underwent laboratory tests including leukocytosis (18,000 mm), platelet (441,000), hemoglobin (10.7), C-reactive protein (3+), normal liver and kidney function tests, and normal coagulate profile tests. Her abdominal ultrasound images showed uterus didelphys with a mixed-pattern cystic mass measuring 72 65 62 mm close to the right ovary plus a solid cystic mass measuring 100 58 mm in the right adnexa. The right kidney was not observed.
We decided to do abdominal and pelvic computed tomography scan to evaluate the cystic mass which was reported in abdominal ultrasound imaging. We had planned to do the surgery in an appropriate condition. This meant with patient's intestinal preparation after evaluating computed tomography results, and in the presence of an expert general surgeon. However, a few hours after the admission, we had to do an emergency surgery on the same night because she had a high fever, increased ESR and CRP, and a possible risk of septic shock. We administered many antibiotics before the emergency surgery because of the acute abdominal pain.
During the first surgery, we did a physical examination while she was under anesthesia. The hymen was normal and intact. But a buldge was found in the right side of her vagina during the rectoabdominal examination. This was confirmed by vaginal examination with a speculum. The right longitudinal septum was revealed with a closed end. At first, we cut the closed end of the hemilateral obstruction of the vagina and then the vaginal septum was resected with an electrocautery device. Afterward, a 200-cc pus was drained out, and this sample was sent to a laboratory for culture and antibiogram.
The patient's abdomen was opened with a midline incision. There was a severe adhesion of the intestine and omentum to the abdominal wall. Since no intestinal preparation had been done and there was a high risk of intestinal rupture, the general surgeon decided that it would be better to end this surgery and prepare the patient for another abdominal surgery in better conditions.
Abdominopelvic-computed tomography scan (with and without contrast which was done after the first surgery) confirmed the ultrasound imaging findings of uterus didelphys with multi-cystic mass with severe development in the right adnexa. A remarkable right hematocolpus hematosalpynx was seen that was due to longitudinal vaginal septum and hemivaginal obstruction. The right kidney was not observed (Figures 1, 2). Her problem was detected as HWW syndrome infected with pyocolpos.
We did the second laparotomy in less than 48 hr after the first surgery with the same incision in the presence of an expert surgeon. Our patient had severe intestinal adhesion and perforated appendix fistulated to the right fallopian tube beside distal small intestinal obstruction. We saw pelvic inflammatory mass beside endometriosis. We did an appendectomy, right salpingectomy, entrolysis, and abscess drainage. Cefepim (Maxipim) and cloxacillin (Jaber-ibn-Hayyan pharmaceutical co) were administrated intravenously as specific infectious disease drugs until seven days after the surgery according to the specimen culture. We ordered continuous low-dosage contraceptive drugs for preventing the progression of endometriosis. Our patient was discharged from the hospital after seven days with a good condition.


This patient is a 57-year-old male with a past medical history of tuberculosis and a significant social history of chronic cocaine use who presented to the emergency department due to progressively worsening dyspnea over the last three months. On physical exam, the patient had diffuse wheezing. He also endorsed a 20-pound weight loss over the last three months and increased urinary retention. The patient denied fevers, chills, recent travel, hemoptysis, and cigarette smoking.
During his hospital stay, the following labs were performed: Acid-fast bacilli smear, Mycobacterium polymerase chain reaction, Legionella antigen, urine drug screen, immunoglobulin E (IgE) levels, Aspergillus antigen, and neutrophilic cytoplasmic antibody. Acid-fast bacilli testing was negative, as well as mycobacterium polymerase chain reaction, Legionella antigen, and neutrophilic cytoplasmic antibody. Urine drug screen was positive for cocaine. Eosinophil count was elevated at 670 cells/μL (normal < 610 cells/μL). IgE level was also elevated at 2,378 kU/L (normal < 114 kU/L). Based on findings of elevated IgE levels, IgE against Aspergillus fumigatus was sought out and came back positive (0.56 kU/L).
Chest X-ray revealed hyperinflation, flattened diaphragm, and pleural thickening (). Chest CT without contrast revealed some bronchial wall thickening and bronchiectasis in the bilateral lower lobes (). In the right upper lobe, there was also some bronchial wall thickening, bronchiectasis, and tree-in-bud nodularity (). A 1.0 × 2.2 × 1.8 cm solid nodule was visualized in the posterior medial right lung (). This was grossly stable in size from a chest CT 3 months prior.
The patient was first treated at an outside hospital for pneumonia with amoxicillin-clavulanate and a 7-day course of prednisone 10 mg at the first onset of his symptoms. He reported transient improvement with steroids. On presentation, prior to confirmation of ABPA, the patient was treated with 60 mg intravenous methylprednisolone BID and inhaled ipratropium bromide with albuterol sulfate. Once ABPA was confirmed, the patient was transitioned from methylprednisolone to a 4-day treatment course with 60 mg oral prednisone. The patient noted significant improvement prior to discharge.
Given the concern for malignancy due to significant weight loss and urinary retention, the prostate-specific antigen (PSA) was measured. This was elevated at 11.2 ng/ml (normal < 4.0 ng/ml). Urology was consulted, and outpatient follow-up with urology was recommended for a transrectal ultrasound- (TRUS-) guided biopsy.


A Japanese boy was born at 30 weeks of gestation by elective cesarean section to a 31-year-old gravida 1 para 0 mother. He was born with an APGAR score of 4, 8 and 8 at 1, 5, and 10 min, respectively, weighing 1,490 g. He immediately breathed spontaneously but showed chest wall retraction. He was provided with a continuous positive airway pressure (CPAP) mask and was admitted to our neonatal intensive care unit (NICU).
His first several days in the NICU were uneventful, and he could tolerate breast milk. On his 7th day of life, he presented with lethargy, bilious gastric residual, and bloody stool. His laboratory results revealed elevated C-reactive protein (CRP) (0.57 mg/dL) and white blood cell (WBC) count (10,630/μL). Chest and abdominal X-ray showed no abnormal signs. Considering the possibility of sepsis, treatment with intravenous ampicillin (200 mg/kg/day) and cefotaxime (200 mg/kg/day) was initiated. His bloody stool resolved quickly, and gastric residuals decreased and then disappeared. On his 8th day, enteral feeding was resumed. His general conditions were stable, his laboratory findings apparently improved, and we completed antibiotics treatment on his 12th day of life. He was tolerant of a feeding increase, and his growth was also stable. Ventilatory support (nasal CPAP and a high-flow nasal cannula) was required until the 15th day of his life.
However, from his 30th day, he presented with increasing apnea and vomiting. Contrast gastric X-ray performed on the 40th day revealed gastroesophageal reflux (GER). Treatment of GER was added by dividing enteral feeding and medication. Nevertheless, his quantity of stool gradually decreased, and physical examination showed worsening abdominal distention. Simultaneously, his CRP and WBC levels were slightly elevated up to his 40th day at 2.56 mg/dL and 20,380/μL, respectively. Intravenous cefazolin (100 mg/kg/day) was administered, followed by cefaclor p.o. Abdominal radiography on his 50th day showed a significantly distended intestine with gas (Fig. ), which urged us to perform contrast X-ray of the colon; the X-ray suggested stenosis of the ascending colon (Fig. ). In order to decompress the distended bowel, we tried to insert a tube through the narrowed colon resulting in failure. Therefore we decided to perform emergent exploratory laparotomy.
Intraoperative findings included viable loops of bowel, and the terminal ileum remarkably distended in the right lower quadrant (Fig. ). The appendix was adhered to the ascending colon by approximately 1 cm from the ileocecal junction and was found to be perforated at its root. This part of the colon was narrow and was not dilated by compression from the oral end, while the contents passed through smoothly. Ascites was slightly yellowish and was examined by a culture test, which turned out to be negative. No perforation in the colon or evident peritonitis was observed macroscopically. The ascending colon lesion including the appendix was resected by 3 cm, and then the ileum and remaining ascending colon were anastomosed end to end.
The pathological findings were as follows: The appendix was perforated at a 2.5-mm diameter 5 mm from the root (Fig. ). The mucosa showed lymphoid hypoplasia. Necrotic granulation tissue replaced the area around the perforation. Old hemorrhage, subtle calcification, and a few foreign body granulomas were found in the area; a finding that is compatible with several weeks of duration of the lesion (Fig. ). By contrast, reconstruction of the luminal layers by fibrosis, suggesting months’ delay, was not observed.
Mucosal erosion of 18 mm in diameter was located on the ascending colon 7 mm from the ileocecal valve in the severely stenotic area (Fig. ). The erosion was a mucosal defect replaced by necrotic granulation, limited within the lamina muscularis mucosae and submucosa. The submucosal granulation and fibrosis are compatible with several weeks of duration. (Fig. ).
Postoperative recovery was good. Enteral feeding was resumed on postoperative day (POD) 4. He was tolerant of full feeding on POD 10. After surgery, he did not develop apnea and his GER treatment was completed. On POD 28, he weighed 3,040 g at the time of his discharge to home. Furthermore, 4 months after discharge, he developed adherent ileus and required surgical detachment. Currently, he is generally well and his neurological development is within the normal range.


A 33-year-old female nonsmoker with a history of breast cancer status post bilateral mastectomies with immediate tissue expander reconstruction presented for bilateral 3D nipple tattooing several months after having her tissue expanders exchanged for silicone implants. The patient did report allergies to penicillin, sulfa, and gluten. At her initial consultation, a “scratch test” was performed using several tattoo inks in the postauricular hairline area to assess for any allergic reaction. The patient did not develop any reaction to the tattoo ink. She returned to clinic 7 weeks after her initial “scratch test” for her definitive tattooing. At that time, NACs were traced on the breasts in appropriate anatomic position and the marked areas were anesthetized with 1% lidocaine with 1:100,000 epinephrine. A Permark coil tattoo device (PMT Corporation, Chanhassen, Minn.) with Electra Pro tattoo ink (Unimax, New York, NY) in 5 colors (brown beige, terra cotta red, chocolate brown, pink beige, cream beige) were used for the 3D tattoo. The procedure was uncomplicated and was tolerated well by the patient. Aquaphor (Beiersdorf Inc., Hamburg, Germany) and Adaptic (Systagenix, San Antonio, Tex.) were applied over the NACs after the procedure. About 7 weeks after nipple tattooing, the patient presented to the clinic with report of an erythematous, pruritic rash around both of her tattooed NACs, which began about 4 days before presentation (Fig. ). She denied any fevers or other infectious symptoms. Her primary care provider had prescribed topical cortisone, and she had been taking Benadryl (diphenhydramine) with little improvement in her symptoms. She was then prescribed a Medrol DosePak (methylprednisolone, 4 mg). Shortly thereafter, her symptoms began to improve and her rash resolved (Fig. ). After completion of the Medrol DosePak, the rash completely resolved (Fig. ).