We report the case of a 24-year-old female who had undergone RYHJ reconstruction 3 years back for choledochal cyst. She presented to us with episodes of abdominal pain localised in the right upper quadrant, associated with fever, itching and jaundice. Physical examination revealed tenderness on deep palpation in the right hypochondrium, temperature of 101.4°F and heart rate 110 beats/min, with negative peritoneal signs. Laboratory analyses were as follows: haemoglobin 9.8 g%, leucocytes 19.99 × 103/UL, neutrophils 93.8%, total bilirubin 7.28 mg/dl, direct bilirubin 6.37/dl, alanine transaminase 76U/L, aspartate transaminase 95U/L, alkaline phosphatase 571U/L, platelet 247 × 103/μl and international ratio 1.6 and negative viral hepatitis panel. Magnetic resonance cholangiopancreatography revealed bilateral hepatic duct dilatation with multiple calculi with anastomotic stricture [].
Owing to the history of RYHJ, we decided to perform LATEM of intrahepatic calculi and anastomotic stricture under general anaesthesia using the standard laparoscopic approach with three trocars. An optical trocar was placed in the midline in between the umbilicus and pubic symphysis and the other two 5-mm trocars at the right and left midclavicular lines just below the umbilicus. The operation began with thorough diagnostic laparoscopy, and after gentle bowel adhesiolysis, the afferent limb, efferent limb and the jejunojejunal (JJ) anastomosis were identified. The right 5-mm port was then extended, and the biliary limb of the jejunum was drawn up to the abdomen. A 1-cm enterotomy was performed on the antimesenteric side of the externalised jejunum, and a side-viewing endoscope (Olympus TJF-Q180V) was inserted into the enterotomy and advanced to the level of the hepaticojejunostomy. A calculus was seen stuck in the anastomotic site which was removed with a biopsy forceps [].
Under portable C-arm fluoroscopy, the right intrahepatic duct was cannulated and cholangiography was performed, which showed evident dilatation of the right intrahepatic bile ducts with multiple stones []. The anastomotic site was dilated up to 12 mm using wire-guided controlled radial expansion balloon (Boston Scientific) at 3 atmospheric pressure, and cholangiolithiasis was cleared using Dormia basket and a Fogarty balloon catheter. The same was repeated on the left hepatic duct obtaining a subsequent normal cholangiogram. The enterotomy was repaired extracorporeally in double layer, the bowel returned to the abdomen and a non-suction tube drain was inserted into the peritoneal cavity. The post-operative period was uneventful, and the patient was discharged 5 days later.


A 72-year-old female patient was hospitalized on September 20, 2018 with stage IV gallbladder cancer with massive intrahepatic and the lymph node metastases in the hilar area and around the head of the pancreas. There was involvement of the duodenum that caused upper gastrointestinal obstruction. Biopsy showed poorly differentiated adenocarcinoma. The patient was extremely exhausted, accompanied by severe anemia with hemoglobin as low as 37 g/L and severe hypoalbuminemia with albumin as low as 23 g/L. The patient had a history of rheumatic heart disease with recent cardiac failure, and fasting blood glucose had been elevated (up to 22 mM) for a long time. Blood tumor markers CA19-9, alpha-fetoprotein, and carcinoembryonic antigen were increased. In addition to receiving symptomatic and supportive care, any conventional “anti-cancer” treatment could not be used. Beginning on October 23, 2018 the patient volunteered to inhale hydrogen-oxygen mixed gas only through a nasal cannula, two hours per day initially, and gradually increased to 6 hours per day. After one month of hydrogen inhalation, the patient showed obvious pseudo-progression of the disease and quickly relieved after symptomatic treatments. After 2 months of hydrogen inhalation, the patient's performance status showed significant improvement (decreased from 5 to 3). After 3 months of hydrogen inhalation, tumor markers were reduced to the normal reference range, and CT showed more than a 50% reduction in gallbladder tumors and multiple intrahepatic and hilar metastases (defined as partial response). The patient's blood sugar dropped to normal when insulin was no longer used. The patient was discharged on February 1, 2019. Currently, the patient continues to inhale hydrogen every day at home and is in the process of further improvement (recent performance status of 0).


A 44-year-old woman diagnosed with lung cancer with multiple metastases in November 2015. Oral targeted drugs were initiated after removal of brain metastases, and most lesions remained stable for 28 months. In March 2018, intracranial multiple metastases, as well as hydrocephalus accumulation in the third ventricle and lateral ventricles, and metastases in bone, adrenal gland, liver were noted. Hydrogen-gas monotherapy was started to control the tumor a month later. After 4 months, the size of multiple brain tumors was reduced significantly, and the amount of hydrocephalus in the third ventricle and lateral ventricles reduced significantly. After 1 year, all brain tumors had disappeared, and there were no significant changes in metastases in the liver and lung. These data show that, after standard treatments had failed, hydrogen-gas monotherapy elicited significant effective control of tumors (especially those in the brain), and survival time was lengthened.


A 53-year-old man reported to the emergency room with a 2-day history of sudden-onset left chest pain radiating to his left back. The pain was intermittent over the 48 h and felt similar to the pain he experienced in a prior episode of pleurisy. It did not localize to any specific region of the chest and did not involve the left arm. It was partially relieved with NSAID use and resting on his left side. The patient had a 30 pack year smoking history as well as a history of illicit drug use. He had history of hypertension that was controlled with lifestyle changes and did not show any signs of end organ damage. The previous clinic visits showed his blood pressure to be under control and on admission it was 100/78. A review of systems and physical examination did not show any signs of paresthesias, numbness, weakness, or ataxia. Cardiovascular, respiratory, and neurologic physical exams were normal. The patient was given nitroglycerin and fentanyl, which eased the pain. Because of both his history of smoking and hypertension as well as his clinical presentation, a chest X-ray, EKG, and blood work-up including troponin I and D-dimer were performed which returned normal results.
Due to high suspicion for coronary ischemia and other cardiac etiologies related to his history of smoking, hypertension, and illicit drug use, the patient was admitted to further investigate his chest pain. At this point, his pain was completely resolved with fentanyl. As part of expanding the differential diagnosis for chest pain, to rule out pulmonary embolism and aortic dissection, a CT angiogram of the chest was performed and revealed a 4.4 × 2 × 2.1 cm soft-tissue mass compressing the spinal cord at the level of T8-T9. To further characterize the spinal lesion, MRI imaging was obtained and confirmed the presence of a heterogeneous mass at the left T8 that was hyperintense on T2-weighted and hypointense on T1-weighted images []. Axial scans showed an intradural extramedullary dumbbell-shaped tumor, characteristic of a spinal schwannoma, at the level of spinal nerve T8.
Given the significant mass effect of the mid-thoracic spinal cord and pain symptoms, the patient underwent an open laminectomy and partial facetectomy of T8 and T9. A dark, dumbbell-shaped mass could be seen extending from the left spinal column grossly []. There also appeared to be a hemorrhage within the dural sac near the T8 nerve root. GTR of the lesion was accomplished with sparing of the nerve root. Post-operative histological examination showed fascicles and nests of plump spindle cells, consistent with schwannoma []. Variable amounts of melanin were also seen within tumor cells [], leading to further immunohistochemical staining. Tumor sections showed positive expression of S100, SOX10, HMB-45, and MART-1 [], compatible with a diagnosis of MS.
A complete history and review of systems did not reveal any family history or clinical signs of Carney complex in this patient. He was discharged 1 day after surgery. We followed up with the patient 2 months after surgery. He was doing well other than some persisting incisional pain. Imaging did not show any local recurrence of the tumor []. He was counseled on his options regarding radiation and referred to another institution for a second opinion due to the rarity of this diagnosis. He has subsequently been lost to follow-up 6 months after surgery.


In June 2014, a 5-year-old boy presented with a 2-month history of fever, night sweats, weight loss, and cough. Physical examination revealed bilateral anterior cervical lymph nodes enlargement which were fixed and painless. The patient also had peripheral cyanosis in digits. Complete blood count test showed hemoglobin concentration: 128 g/L, white blood cell count: 9.12×109/L (30.1% neutrophils, 57.3% lymphocytes, 6.61% eosinophils, 4.54% monocytes, 1.45% basophils) and platelet count: 329×109/L. Liver enzymes, serum bilirubin, creatinine, and blood urea nitrogen were all normal. Blood culture showed no bacterial growth.
Chest X-ray revealed bilateral airway narrowing. Neck computed tomography (CT) scan showed multiple bilateral anterior cervical as well as supraclavicular and infraclavicular lymphadenopathy causing mass effect. The largest lymph node measured was 20 × 15 mm. Some lymph nodes showed cystic degeneration suggestive of central necrosis. The thyroid gland exhibited heterogenous density. CT scan of the chest demonstrated bilateral nodular and micronodular lung densities and hilar lymphadenopathy [Figures–]. Abdominal CT scan showed mild hepatomegaly and paraaortic lymph node enlargement. The overall clinical impression was favoring either tuberculosis or Hodgkin’s lymphoma (HL).
Based on the clinical suspicion of lymphoma, an excisional biopsy of a single cervical lymph node was performed. A bone marrow trephine biopsy was also obtained for staging. Histopathological examination of the lymph node showed proliferation of malignant thyroid glands, demonstrating papillary growth and characteristic nuclear clearing and grooves of follicular epithelial cells. Only a residual rim of lymph node tissue was present [Figures –]. The diagnosis of metastatic papillary thyroid carcinoma was established. The bone marrow biopsy was negative for metastasis. The patient was in clinical stage-I disease.
The patient was transferred to another facility and had total thyroidectomy and cervical lymph nodes excision. Then he received radioactive iodine therapy. The patient remained in complete remission until December 2019 when he developed respiratory symptoms. CT scan revealed multiple bilateral pulmonary nodules consistent with metastasis. The patient had many admissions to the intensive care unit for unstable vital signs. He died in March 2020 of septicemia by methicillin-resistant Staphylococcus aureus.


A 43-year-old man presented in 2015 with inflammatory back pain for 3 years. The patient’s erythrocyte sedimentation rate (ESR) was 100 mm/hour and C-reactive protein (CRP) was 130 mg/l. Spine and pelvic MRI showed bilateral active sacroiliitis.
The diagnosis of spondyloarthritis, in its axial form, was established according to the Assessment of SpondyloArthritis International Society (ASAS) criteria.
The patient received NSAID for one year but finally without satisfactory clinical improvement according to the Bath Ankylosing Spondylitis Disease Activity Index (BASDAI) criteria and therefore was treated with adalimumab for two and a half years with complete remission.
In January 2019, the patient presented a transient ischemic attack with a normal cerebral angioscan. On admission, the patient was asymptomatic and the history gave no clue of an ongoing vasculitis. The clinical examination revealed comparable normal blood pressure values in both arms, and slightly decreased radial and pedal pulses. The pulse in the extremities was hardly discernible, with presence of a bilateral umbilical bruit, bilateral carotid bruit and a right subclavian artery bruit.
The thoracic angio-CT showed the presence of multiple significant stenoses of the supraaortic arteries (right subclavian artery () and both vertebral arteries, the thoracic and abdominal aorta. The digestive tract arteries (superior and inferior mesenteric artery) and both renal arteries were also constricted (), with celiac trunk stenosis of 60%.
The laboratory examinations showed elevated markers of inflammation (CRP: 70 mg/l, ESR: 55 mm/h). The diagnosis of TA was retained and it was stage 5 according to the Tokyo 1994 classification.
Adalimumab was stopped and the patient underwent a 3-day regimen of methylprednisolone (15 mg/kg/day) followed by a 1 mg/kg/day prednisone dose. Methotrexate (MTX) was maintained at the dose of 25 mg p.o. weekly. Under this treatment, a good response with normalization of the inflammatory parameters was observed.


shows the electrocardiogram (ECG) of a 24-year-old boy with an overt preexcitation due to a right-sided posteroseptal accessory pathway (delta wave positive in I, II, and aVL, V2–V6 and negative in III, aVR, and V1). The patient was referred to our center because of recurrent episodes of palpitations. The accessory pathway was identified in the right posteroseptal portion of the right atrium, near the ostium of the coronary sinus, and a radiofrequency application successfully resulted in disappearance of the preexcitation and dissociation of ventriculo-atrial retroconduction. The ECG performed 20 h later still showed the absence of the prexcitation and also deep, giant, negative T-waves in II, III, and aVF [], secondary to “cardiac memory.”


We obtained written informed consent from the patient after surgery to publish this report.
A 60-year-old female presented with chronic pain in the right ankle and foot. She had right foot ligament tear, which included anterior talofibular ligament, calcaneoufibular ligament and posterior talofibular ligament, followed by a modified Brostrom surgery on the right foot in March 2017. However, the patient still felt a painful sensation, the numerical rating scale (NRS) was 10 of 10, at the right foot. She also complained of allodynia, burning and tingling sensation, leg edema, and change of skin color with a dystonic posture of the right foot. The result of electromyography and nerve conduction velocity was no abnormal findings. However, bone scan showed diffusely decreased perfusion, blood pool and bone uptake of right foot and quantitative sudomotor axon reflex test suggest sympathetic postganglionic sudomotor dysfunction or sweat gland abnormality in right distal leg. Department of orthopedic surgery in our institution gave an opinion that there were no joint pathology, bony abnormality and nothing to cause the pain with orthopedic opinions. Following a clinical assessment, she was diagnosed with CRPS type 1 according to the International Association for the Study of Pain clinical diagnostic criteria []. Although she was treated with several medications, physical therapy, lumbar epidural blocks, and lumbar sympathetic ganglion blocks, her symptoms remained intractable.
In August 2018, she was considered for SCS trial. During the SCS trial, strict aseptic techniques and prophylactic antibiotics (cefazolin) were used. A guide needle for an electrode was inserted through the right L1-2 paramedian epidural space, and the tip of the electrode was placed at the T9-10 level. After the induced paresthesia was confirmed at her painful sites, the lead was fixed. During a test period, her pain and allodynia decreased from 9 to 0 on NRS. We implanted a permanent implantable pulse generator (IPG) in a subcutaneous pocket of her left lower abdomen.
On the fifth postoperative day, the patient had back wound dehiscence and pus discharge back incision site. The neurological examination was normal. Computed tomography (CT) and thoracic MRI showed no abnormality except a back skin wound. The culture report of the specimen of the back wound discharge revealed extended-spectrum β-lactamase (ESBL) producing Escherichia coli. Following a discussion with the Department of Infectious Disease in our institution, ertapenem was immediately started and the patient underwent surgical wound exploration under general anesthesia. The wound problem was resolved; hence, she was discharged on ciprofloxacin and followed-up at the out-patient clinic. However, wound dehiscence at the back incision site was observed again after 2 months. Although a plastic surgeon performed wound revision with local flap twice, the wound problem did not resolve, so we decided to remove the SCS system.
In March 2019, we consulted the Department of Infectious Disease in our institution and found that there was no evidence of residual infection. The patient was treated through oral medication, but complained the same symptom as before. Therefore, we decided to re-insert the SCS electrode. The electrode insertion was planned through the contralateral T12-L1 epidural space to evade the previous infection site. However, during the lead insertion, it was impossible to advance up to the T11 level of the epidural space. Moreover, the contrast dye did not spread above the T11 level of the epidural space. Consequently, we concluded that the epidural adhesion was interrupting the electrode advance. We supposed that epidural adhesiolysis to resolve the epidural adhesion would be helpful for the insertion of the SCS. After epidural adhesiolysis with a Racz catheter, however, the electrode was still not placed up to the T11 level (). Therefore, we additionally tried to perform the combined epidural balloon decompression and adhesiolysis with an inflatable balloon catheter (Zineu®, JUVENUI, Korea) (, ). After combined balloon decompression and adhesiolysis, the lead moved well, and we successfully implanted the SCS electrode at the T9-10 level. After the induced paresthesia was confirmed at her painful sites, the new IPG device was then repositioned in a subcutaneous pocket, on the left lower abdominal region (). Tingling sense decreased from 10 to 2 on NRS, and burning sensation decreased from 10 to 5 on NRS. The intravenous administration of ertapenem to cover ESBL producing E. Coli was continued for a 2 week period, following which the blood culture result was negative. There were no postoperative complications, and the patient was discharged in a good physical state. The symptom of patient has been well controlled with the SCS to date.


A middle-aged man in his 40s was affected by a right cerebellar/brainstem infarction. Before the stroke, he had been employed in the warehouse logistics, an environment requiring high levels of standing balance for operating forklifts. One day, he suddenly fell at his residence and was then rushed to the hospital. On arrival, consciousness disturbance, persistent gaze deviation, left haemiplegia and right cerebellar ataxia were noted. Diffusion-weighted cranial MRI performed the same day revealed an infarct in the right cerebellum and brainstem (). Following acute treatment and care, the patient was transferred to our hospital on the 37th day after onset for recovery-phase rehabilitation.


A 15-year-old female patient was referred to us for evaluation of uveitis treated in other clinics before. Child reported first symptoms 5 years ago, which consisted of blurry vision in right eye first then left eye and redness of eyes. She had oral aphtae and oral ulcers as well, that improved over time with treatment. She was treated earlier for right sided knee inflammation. She has been diagnosed with uveitis and for 5 years treated with on and off topical, peribulbar and systemic corticosteroids with periods of improvement and periods of worsening of symptoms. She developed cataract in both eyes and was planned for surgery two years ago, but parents refused surgical intervention. Two years prior to referral to our clinic she had laser fotocoagulation in both eyes due to retinal vasculitis and was started on Methotrexat 15 mg weekly. Despite the administration of local and systemic corticosteroids and MTX, inflammation persisted; therefore, the patient was referred to our clinic for second opinion.
The best corrected visual acuity (BCVA) was 0.9 in RE and 0.6 in LE, and the IOPs of the right and left eye were 17 and 18 mmHg, respectively. Anterior chamber cell grade of 0.5+ in the both eyes. She had posterior synechiae in both eyes and posterior cortical cataract in both eyes (). The vitreous cell grade was 3+ in the right eye and 2+ in the left eye. On ultrasound she had inflammatory reaction in vitreus, more prominent in right eye (). Posterior segment was hazy due to vitreal changes and cataract, but laser burns could be seen as well as signs of vasculitis and macular changes. We performed fluorescein angiography and SD OCT on patient to reveal intraretinal cysts on both eyes and perifoveolar oedema right eye ( and ).
Non-ocular manifestations oral ulcers were improved with previous treatment. Skin or genital lesions were not observed. She was refered to pediatric rheumatologist for complete evaluation. All laboratory workup was within normal limits. Chest X ray and abdominal ultrasound were normal. Interferon-gamma release assay and Quanti Feron Tb gold test for tuberculosis infection and Treponema pallidum antibody hemagglutination test for syphilis were negative; Hepatitis HBs and anti HBc were negative as well as HCV Ag; angiotensin-converting enzyme, antinuclear antibody, and anti-citrullinated protein antibody levels were within the normal range. MRI of brain and cervical spine were normal, as well as EEG. Neurological examination was normal. She tested positive for HLA-B51. There was no family history of autoimmune diseases. Following treatment, initiated in the previous clinic, was continued: 0.1% dexamethasone eye drops (four times/day), tropicamide/phenylephrine eye drops (once/day), Methotrexat (MTX) 15 mg once a week followed by folic acid. The patient was diagnosed with the incomplete type of Behçet’s disease on the basis of the presence of a typical ocular symptoms and recurrent oral ulcers, retinal vasculitis recurred in both eyes in spite of treatment with MTX, therefore, initiation of adalimumab was proposed. Subcutaneous adalimumab injection was then introduced. The patient was 15-year-old and weighed 57 kg when adalimumab was initiated, thus we administered 40 mg every 2 weeks. After 2 months BCVA improved in both eyes, to 1.0 RE and 0.7 LE. The anterior chamber in both eyes improved to 0 , within 2 months after beginning adalimumab administration. Posterior synechiae and cataract did not change. Oral ulcers and problems with knee did not repeat after adalimumab therapy. The inflammation subsided. After three months she was taken off local medication. On the first follow up SD OCT showed significant improvement () as well as after 18 months.
After 12 months with adalimumab we decided to lower MTX dosage to 10 mg once per week and she remained symptoms free with this lower dose. The follow-up period after introduction of adalimumab was 18 months (at the time this report was written) and the patient did not experience any side effects. Her ocular and oral symptoms of BD were well controlled.


A 37-year-old man with deafness and vision impairment because of retinitis pigmentosa since childhood and a MYO7A gene mutation denoting Type 1 US, was admitted because of recurrent syncope. Blood pressure was normal, and physical examination was unremarkable except for irregular heart rhythm. ECG showed sinus rhythm 80 bts/min and frequent ventricular extrasystoles with right bundle branch block configuration often in ventricular bigeminism. At Holter monitoring 15 280 ventricular ectopic beats were registered in 24 h including couples, triplets, and short phases of non-sustained ventricular tachycardia.
Cardiac magnetic resonance (CMR) images showed a mildly dilated left ventricle (End diastolic volume [EDV]/Body surface area [BSA]: 105 mL/m2) with normal thickness of ventricular wall (Figure
), and moderate reduction of systolic function (EF: 42%, Figure
).
Interestingly, the patient presented systolic mitral-annular disjunction with mitral valve prolapsed and moderate valve regurgitation (see arrow, Figure
). Mitral annular disjunction occurred with hypotonia of posterior papillary muscle and abnormal stretching of the cords (Supporting information, and ) suggesting contractile impairment. No areas of myocardial fibrosis were detected at late gadolinium enhanced imaging (Figure
). No CMR criteria for left ventricular myocardial non-compaction were recognized at short axis view (Figure
). Right ventricular morphology, size, and systolic function were normal. To investigate cause and pathway of cardiomyopathy, after informed consent, the patient underwent cardiac catheterization with coronary and left ventricular (LV) angiography and endomyocardial biopsy (EMB). Coronary arteries were normal.


A 65-year-old male presented with a chief complaint of ulcer on the right lateral aspect of the tongue for 1 year []. History of present illness revealed a small-sized ulcer in the initial phase of peanut shape which is gradually reached to the present size. The pain was moderate, continuous and radiating to the cheek and neck region which did not relieve after medication. The patient did give a history of local trauma by adjacent teeth. On clinical examination, an ulcer was seen on the right lateral border of the tongue, opposite teeth 45 and 46. Close inspection revealed an erythematous ulcer of 2 cm diameter with a whitish surrounding halo. On palpation, the ulcer was smooth, tender and firm in consistency, with well-defined margins among everted rolled and induration. There was no fixation to the deeper structures. Submandibular lymph nodes were tender and palpable, while deeper cervical lymph nodes were nonpalpable. Provisional diagnosis of traumatic ulcer was made. An excisional biopsy was performed under local anesthesia for histopathological examination.
The possibility of malignancy was ruled out by ultrasonography. Microscopic examination revealed under scanner view/low-power view, two pieces of tissue showing irregular ulcerated epithelium overlying intensely inflamed connective tissue stroma. The deeper stroma shows a group of muscles fiber bundles with some areas showing inflammatory cell infiltration []. Under high-power view, it reveals that epithelium is stratified squamous parakeratinized type. The connective tissue stroma depicts loose to dense bundles of collagen fibers with plump to spindle-shaped fibroblasts. Intense chronic inflammatory infiltrate predominantly comprising lymphocytes, eosinophils and macrophages/histiocytes is evident. The inflammatory components appear to infiltrate deep into the underlying muscle fiber bundles. In some areas, mild degenerative changes are also absorbed in muscle fibers [Figures and ]. The clinical differential diagnosis may include traumatic neuroma, oral lymphangioma atypical histiocytic granuloma. A traumatic neuroma is a type of neuroma which results from trauma to a nerve, usually during a surgical procedure. The most common oral locations are on the tongue and near the mental foramen of the mouth. The lesion clinically visualized as TUGSE.
Oral lesions frequently appear on the tongue showing irregular nodularity demonstrating pebbly appearance and translucent hue owing to its superficial location and having gray, pink or yellowish color, which is also a pathognomonic feature of lymphangioma of the tongue. The lesions of atypical histiocytic granuloma are clinically similar to squamous cell carcinomas or specific granulomatous ulcerations. Several therapeutic approaches such as topical steroids, mouthwashes, topical antibiotics, curettage and cryotherapy have been reported.[] The most frequently performed therapy is simple surgical excision. No local recurrences are usually noted after excision.[] In our case, simple excision was performed and a rapid improvement of the ulcer was seen similar to what was described by other authors.[]


A 10-year-old female child reported to our unit complaining of a swelling in the left lower jaw region behind the teeth for the past 4 months. Swelling started as a peanut size and increased to the present size. Facial symmetry was noted extraorally, but cervical lymph nodes were not palpable. Intraoral examination revealed a solitary, pedunculated, well-circumscribed, nonfluctuant, nonsuppurative swelling, distal to the lower left first molar tooth extending into the retromolar region measuring 3 cm × 3 cm approximately in the greatest dimensions. Overlying surface on the superior aspect revealed indentations of upper teeth []. Swelling was nontender without localized rise in temperature. Fine needle aspiration was negative. The permanent mandibular left first molar was slightly mobile. No anesthesia/paresthesia was noticed. No relevant medical history was noticed.
An orthopantomogram revealed that the swelling was not odontogenic in origin. Radiolucent shadow of the soft tissue growth with erosion of bone in distal to first molar and retromolar region was observed []. Based on the clinical and radiographic examination, a provisional diagnosis of fibroma was made. Considering the age and location of the swelling, an excisional biopsy was planned under general anesthesia (GA). Patient written consents were obtained. Following complete systemic evaluation, surgical excision was done under GA. Macroscopically, it is pinkish in color, elastic in consistency, oval in shape and well encapsulated []. No teeth were extracted.
Microscopic findings revealed a stratified squamous epithelium with irregular acanthosis with ulceration covered by neutrophilic exudate and underlying granulation tissue. Subepithelial stroma shows an ill-defined lesion composed of spindle cells arranged in interlacing fascicles and bundles with ill-defined cytoplasm and mildly pleomorphic vesicular nuclei with inconspicuous nucleoli. Interspersed capillaries are seen suggestive of low-grade spindle cell neoplasm []. Immunohistochemistry was done which revealed immunoreactivity for SMA and negativity for the desmin and S-100 protein []. The patient was kept on periodic follow-up without any signs of recurrence.


A 69-year-old male with a history of Stage III immunoglobulin G Lambda multiple myeloma. He had been treated with bortezomib, lenalidomide, and dexamethasone. He had completed 5 cycles of cyclophosphamide, bortezomib, and dexamethasone. Due to the coronavirus disease-2019 pandemic and to prevent exposure to hospital for chemotherapy infusions, he was transitioned to an oral chemotherapy regimen. He was initiated on ixazomib 3 mg, dexamethasone 36 mg, and lenalidomide 25 mg. He was started on a regimen of ixazomib 3 mg to be taken on days 1, 8, and 15; dexamethasone 36 mg on days 1, 8, 15, and 22; and lenalidomide 25 mg on days 1 to 22. The patient, instead of taking ixazomib weekly, took the ixazomib daily for 3 days.
He was admitted with nausea, fatigue, and profuse watery diarrhea. He also had developed a diffuse macular rash in his lower extremities extending to the part of his lower abdomen, which was non-pruritic. Initial blood pressure on arrival was 79/51 mm Hg. He was afebrile, and his pulse was 84 beats per minute. Laboratory studies throughout the hospital course are listed in . He was initiated on a fluid bolus with 2 L and was started on maintenance fluids. His blood pressure improved initially; however, due to worsening of blood pressure, he required transfer to the medical intensive care unit. He was initiated on norepinephrine infusions to maintain mean arterial blood pressure above 65 mm Hg. He was empirically initiated on vancomycin, cefazolin, and metronidazole. A diarrhea panel obtained for several common gastrointestinal pathogens () was negative. A computed tomography image of the abdomen was obtained, which showed scattered regions of mild mural thickening and the presence of mild inflammation in the small bowel loops suggesting possible enteritis. One out of the 2 blood cultures collected at the time of admission returned positive for methicillin-resistant Staphylococcus aureus (MRSA) from the port site. His antibiotics were ultimately changed to daptomycin. He underwent transthoracic and transesophageal echocardiography, which did not show any evidence of vegetation and he completed a course of 2 weeks of intravenous daptomycin for MRSA along with the removal of the port.
Hemodynamic and laboratory parameters improved over the first week of admission; however, the patient continued to have ongoing diarrhea. He initiated on loperamide and diphenoxylate without improvement. General surgery was consulted and determined no surgical intervention was deemed necessary. He was then started on octreotide with improvement in diarrhea. He was ultimately discharged in stable condition to a subacute rehabilitation center. His chemotherapy was not initiated on discharge. His chemotherapy was then initiated 2 months post-discharge and was changed to carfilzomib and dexamethasone and he currently is in stable condition.


A 22-year-old male complained of infertility without any other discomfort.
The couple had been trying to conceive for 2 years.
The patient had an unremarkable medical history.
Personal and family history showed no specific illnesses.
The patients’s physical examination showed no abnormalities. His blood pressure (BP) and heart rate (HR) were 17.3/9.3 mmHg and 80 bpm, respectively.
The levels of catecholamines in blood (norepinephrine 283.1 pg/mL, epinephrine 47.5 pg/mL, dopamine /) and urine (norepinephrine 93 μg/24 h, epinephrine 3 μg/24 h, dopamine 137 μg/24 h) confirmed that the adrenal gland masses were pheochromocytomas.
Computed tomography (CT) results indicated the association of multiple lesions: (1) Bilateral adrenal gland mass (Figure ): The size of the left mass was 4.5 cm × 4.3 cm × 4.5 cm, and the right mass was 2.6 cm × 2.2 m × 3 cm; (2) Cysts in the upper region and a low-density occupying lesion on the left kidney; (3) Cystadenomas of the pancreas; and (4) Space occupying lesion with supply blood on the epididymis.
The patient was advised to undergo extensive imaging examinations. Multiple cystic nodules were detected in the thyroid by ultrasound. A single hemangioblastoma in the brainstem was characterized by magnetic resonance imaging, the diameter of which was approximately 5.6 mm along the medulla oblongata, which correlated with the clinical manifestations of Type 2B genotype-phenotype classification in families with VHL syndrome.


A 56-year-old man came to our Ophthalmology Unit complaining of blurred vision, foreign body sensation, burning and itching in his right eye. His best corrected visual acuity (BCVA) was 5/10 in the right eye. The patient did not wear corrective spectacles, and denied trauma and exposure to chemicals.
The slit lamp examination revealed slight lid oedema and conjunctival hyperaemia; and the fluorescein stain test revealed branching epithelial lesions with terminal bulbs consistent with the situation that is classically observed with herpes simplex virus (HSV) epithelial keratitis (shown in ). The corneal stroma was clear, and there was no inflammation in the anterior chamber or posterior segment. The patient’s left eye was normal. Valaciclovir 1000 mg tablets (1 tablet/day) and liposomal ozonated oil eye drops 3 times a day in the right eye were prescribed. Three days later, a decrease in the fluorescent area and dendritic thickness (), accompanied by a reduction in pain, lacrimation and photophobia was observed. By day four, the corneal involvement and fluorescein staining were practically resolved (), and a further reduction in symptoms occurred. After one week of treatment, the corneal examination showed good healing of the defect (), and a full recovery of visual acuity in right eye was achieved.
In this clinical case, adding liposomal ozonated oil to the systemic antiviral therapy significantly shortened the healing time and reduced the clinical symptoms in just 7 days. After the first week, the patient suspended the systemic antiviral treatment, and continued liposomal ozonated oil (3 times a day) alone for 1 month, leading to complete recovery of sight, no signs of corneal opacities and a complete regression of the symptoms.


A 63-year-old woman was hospitalised for a corneal abscess. Three days earlier, due to severe redness and a foreign body sensation, the patient had received topical antibiotic therapy with a rapid improvement in the objective clinical signs and symptoms, which subsequently got worse. At admission, the patient presented lagophthalmos, associated with the inability to close her left eyelid completely (Bell’s palsy), presence of conjunctival hyperaemia, with an accumulation of mucous and purulent secretions, and persistent discharge at the fornices. She also presented severe corneal infection, corneal abscess, and corneal oedema with Descemet’s folds (). Anterior segment OCT images revealed hyper-reflectivity with high scattering effect due to a dense accumulation of abscess material and an increased corneal thickness (). Conjunctival swabs were taken for microbiological investigations, and the patient started therapy with Ceftazidime pentahydrate for injection, 2 g/vial (1 vial/day), Atropine 1% eye drops (2/day), Levofloxacin eye drops (hourly), Gentamicin eye drops (hourly), and liposomal ozonated oil eye drops (5/day). After 3 days of treatment, the objective clinical situation showed an initial improvement, with a reduction of abscess area, corneal oedema and corneal infection (). After 5 days, a further improvement was seen, and initial draining of the abscess was performed ().
Systemic therapy with Acyclovir 400 mg tablets, 5 times a day for 5 days was prescribed. Corneal sensitivity was qualitatively evaluated with a wisp of the cotton-tipped applicator; complete anaesthesia was observed in all 4 quadrants of the cornea. The presence of palsy, lagophthalmos, inability to close the left eyelid completely, eye pain and the corneal patterns at confocal microscopy suggested the corneal abscess had a neurotrophic aetiology. Acyclovir was continued at 400 mg twice-daily for 15 days, then 400 mg/day for 1 month. The confocal microscopy revealed the presence of severe inflammatory cell infiltrate, which almost completely replaced the keratocytes, caused by the viral agent (). Clusters of round inflammatory cells and corneal nerve fibre damage were evident, as neurotrophic keratopathy can exacerbate autoimmune post-herpetic stromal keratitis.
Therapy with liposomal ozonated oil (3 times a day) was continued, and after 7 days, a significant reduction was seen in the affected area on the fluorescein test (). Ten days later, restoration of epithelial integrity was observed ( and ) with a slight positive fluorescein test. The OCT showed a gradual and progressive normalisation of corneal anatomy, with resolution of the abscess area and ulcer (, and ).


A 37-year-old woman was referred to our hospital for evaluation of elevated liver enzymes and hematuria. The patient provided verbal informed consent for the publication of this case and accompanying images. She did not have a history of smoking or alcohol use disorder. She did not use medications and was not exposed to toxic materials. At the time of admission, she complained of fatigue and dark urine and denied any family history of liver or hematological diseases. A physical examination subsequently revealed that she had an anemic appearance.
One month earlier, the patient had been diagnosed with PBC and a urinary tract infection at another hospital on the basis of the presence of the M2 fraction of AMA (AMA-M2) and elevated immunoglobulin (Ig) M, gamma-glutamyl transpeptidase, and alkaline phosphatase levels. Additionally, aspartate aminotransferase (AST) and alanine aminotransferase (ALT) levels were slightly elevated (130.7 U/L and 206.0 U/L, respectively). Abdominal ultrasound and a multi-slice computed tomography (CT) scan of her hepatobiliary area revealed that she had a fatty liver. AIH was not excluded on the basis of positive antinuclear antibodies (ANA; 1:1000) and elevated IgG and r-globulin levels. The patient was treated with 750 mg/day ursodeoxycholic acid (UDCA), which led to an improvement of the symptoms and cholestatic enzyme levels.
Initial urinalysis results revealed 2+ occult blood in the urine, 12 red blood cells (RBCs)/high-power field, and a negative urine culture. Blood test results revealed moderate anemia (RBC count, 2.47 × 1012/L and hemoglobin, 87 g/L) and liver dysfunction (AST, 130.7 U/L; ALT, 206.0 U/L; gamma-glutamyl transpeptidase, 457.7 U/L; and alkaline phosphatase, 619.1 U/L). The globulin levels were as follows: r-globulin, 37 g/L; IgG, 32.6 g/L; and IgM 7.68 g/L. Hematuria position diagnosis revealed mixed hematuria with 30% polymorphic and 70% morphologically uniform erythrocytes.
An extensive viral hepatitis workup, including polymerase chain reaction assays for hepatitis A, B, C, D, E, and G viruses, Epstein–Barr virus, and cytomegalovirus, revealed negative results. Histological examination of the percutaneous liver biopsy specimens revealed nonsuppurative destructive cholangitis and moderate interface hepatitis (), suggesting the overlap of PBC and AIH.
Further investigation revealed cell-free plasma hemoglobin level of 200.5 mg/L and a reticulocyte percentage of 6.62%; the patient had negative serum acid glycerin hemolysis and anti-human globulin test results. There were no findings that suggested iron, folic acid, or vitamin B12 depletion. Bone marrow biopsy revealed hyperplastic anemia without other significant abnormalities (). Flow cytometric analysis of the peripheral blood revealed the following: totally deficient RBCs (type III), 28.45%; partially deficient RBCs (type II), 28.22%; CD24−/fluorescent aerolysin (FLAER)− cells (PNH granulocytes), 87.36% of granulocytes; and CD14−/FLAER− cells (PNH monocytes), 89.07% of monocytes (). These results led to the diagnosis of PNH.
On the basis of the results of the extensive workup, the patient was definitively diagnosed with PBC–AIH overlap syndrome and PNH, and she was treated with 750 mg/day UDCA and 25 mg/day oral prednisolone after she provided consent. The patient showed dramatic improvements in routine urinalysis and liver function test results over the 2 monthly follow-up examinations at the outpatient clinic after treatment was initiated (). Prednisolone was reduced to 15 mg/day. The patient started to control her weight. Seven month later, the patient was symptom-free with a near-normal range of laboratory findings, and there were no indications of a fatty liver. However, the ANA titer results were still 1:1000. Prednisolone was discontinued, and the patient continued treatment with UDCA. At the 24-month follow-up evaluation, the patient had recovered well.


A 55-year-old male presented with multiple hairless patches on his scalp dating back 1 month. He had suffered from an HCC and had been treated with nivolumab for 6 months after a hepatectomy. Five months after taking nivolumab, patient's hair loss started. He was treated for hair loss with triamcinolone intralesional injections at a different hospital without improvement. A physical examination revealed multiple hairless patches on his entire scalp without eyebrow, eyelash, or other body hair involvement (). Nail pitting was not found. The severity of his alopecia tool (SALT) score was evaluated as grade S2. He didn't have any previous history of AA. We performed skin biopsy on the hairless patches of his scalp. Histopathological findings revealed a decreased number of hair follicles with perifollicular lymphocytic infiltration. Most follicles were in the telogen stage, and the telogen/anagen hair ratio was approximately 1 (). Dermoscopic findings revealed lots of broken hairs with black dots. With clinicopathological correlations, we diagnosed him as having AA. Treatment included topical steroids and minoxidil. Despite 2 months of topical treatments the patient's hair loss was aggravated with resultant alopecia totalis (). His SALT score was upgraded to S4. No hair regrowth was noted after 4 months of follow-up. After his alopecia treatments, the patient was diagnosed with thyroid metastases during his routine follow-ups, and he underwent additional radiotherapy. With this patient's condition, he didn't undergo AA treatment.


A 32-year-old nulliparous woman visited because of atypical squamous cells, to consider high-grade squamous intraepithelial lesions on cervical cytology. Cervical intraepithelial neoplasia III was seen on cervical biopsy; thus, we conducted loop electrical excision procedure, and the pathologic finding demonstrated microinvasive squamous cell carcinoma with a vertical invasion of 1 mm/3 mm in total thickness and horizontal spread of 4 mm without lymphovascular space invasion. To preserve her fertility, cold knife conization was performed again because of a positive resection margin showing high-grade dysplasia. However, the pathologic findings showed invasive squamous cell carcinoma with a vertical invasion of 4.5 mm/10 mm in total thickness and horizontal spread of 8 mm with negative surgical margin on the 12 to 3 o’clock specimen. To establish the clinical stage of cervical cancer, we performed computed tomography (CT) and magnetic resonance imaging (MRI) of the pelvis, which demonstrated no evidence of local tumor invasion or lymph node metastasis. Positron emission tomography-CT showed no definite malignant lesion, and sigmoidoscopy and cystoscopy showed no abnormal findings. Finally, she was diagnosed with stage IB1 cervical cancer, and we proceeded with a robot-assisted uterine artery-preserving radical trachelectomy, considering her strong desire to preserve fertility.
For this surgery, a four-arm da Vinci Si system (Intuitive Surgical, Inc., Sunnyvale, CA, USA) with a standard port placement and a set of robotic instruments for the pelvic surgery were used for the procedure []. In the pelvic cavity, we identified one enlarged lymph node in the left external iliac artery area, which was negative for malignant cells upon frozen section. Then, we performed systematic pelvic lymphadenectomy and para-aortic lymph node sampling until the level of the inferior mesenteric artery. Thereafter, radical trachelectomy preserving the uterine arteries was performed (), and a cerclage was placed with a transvaginal MERSILENE® polyester fiber suture. Uterovaginal reanastomosis was made through the vaginal approach, and a 6fr 2-way Foley catheter was placed in the uterine cavity to maintain patency.
Total operation time was 330 minutes, and blood loss was approximately 200 mL. The final pathological examination showed no residual tumor in a 3.5 cm-sized specimen of the cervix and vagina, and no lymph node metastasis among the 17 resected lymph nodes. Six months after surgery, no abnormal findings were observed (), and she was deemed fit for pregnancy through assisted reproductive technology.
She received clomiphene citrate for five days from the second day of her menstrual cycle, and human menopausal gonadotropin for seven days followed by successful implantation with intrauterine insemination 14 months after surgery. Progesterone intramuscular injection and vaginal tablets were used for implantation support.
As with other mothers, nuchal translucency measurements and pregnancy-associated plasma protein-A levels were checked at 12 weeks of gestation, and a Quad test was performed at 16 weeks of gestation. The integrated test showed low risk. She had no risk factors except for antibiotic treatment with repeated acute pyelonephritis during pregnancy.
She presented with abdominal pain at 21 weeks and three days of gestation, and a 2 cm bulge of amniotic sac was observed on ultrasonography and speculum examination. Thereafter, she was hospitalized and treated with prophylactic antibiotics due to incompetent internal os of the cervix. At 22 weeks and one day of gestation, membrane rupture occurred. At 24 weeks and three days of gestation, uterine contraction occurred and patient was started on tocolytics. Although she had received tocolytics for suppression of uterine contractions, regular contraction and fetal descent proceeded abruptly at 26 weeks and 3 days of gestation. Since the fetus had already descended to stage 3 and there was no fetal heart rate deceleration, we decided to proceed vaginal delivery instead of preparing for cesarean section. Because the fibrotic ring at the level of uterovaginal anastomosis interfered with fetal descent, we removed the cerclage knot and incised the ring with scissors at 2,10 o’clock to facilitate vaginal delivery.
As a result, she immediately delivered a preterm male baby with no anomalies, weighing 830 g, with APGAR scores of 1 and 4 at 1 and 5 minutes, respectively. Umbilical cord PH 7.334 showed no fetal hypoxia. After delivery, the incision wound healed without suture as with the lacerated cervix. Although the baby had been treated at the neonatal intensive care unit, he presented with no complications, and was healthy five years after delivery.


A 47-year-old woman presented with progressive lower back pain for 1 year associated with fecal incontinence for 2 years and urinary incontinence for 1 year. However, no past history of pulmonary tuberculosis and diabetes mellitus was recorded. On examination, no obvious swelling was noted. However, patient complained of local tenderness at sacral region. No cervical lymphadenopathy was recorded. On radiological imaging studies, she had an approximately 6.5 × 5 cm lobulated expansile mass involving S1 and S2 vertebrae. The mass was encroaching sacral canal and foramina, superiorly reaching upto L5- S1 recess. On imaging, diagnosis of chordoma was rendered. All hematological and biochemical parameters were within normal limits. Thyroid hormone assay were also normal. Sagittal MRI scan revealed an extradural mass with spinal canal stenosis []. Core needle biopsy was performed from the mass under guidance. Biopsy revealed follicles/acini arranged in back to back manner. Keeping in mind the architectural pattern differential diagnosis of metastatic adenocarcinoma from breast, colon, etc., and FTC were deliberated on histology alone []. Subsequently, panel of immunohistochemical markers was carried out. These follicles were intensly and diffusely positive for TTF-1 & thyroglobulin []. These follicles were non immunoreactive for CK7 & CK20, respectively. Thus, diagnosis of metastatic follicular carcinoma from thyroid was rendered on histology and immunohistochemistry. Further eliciting the clinical history, patient had history of hemithyroidectomy 6 years back, following which the patient was put on thyroxine. Unfortunately, patient had no histopathology reports available from prior surgery. Subsequently, thyroid ultrasound study revealed a 3 × 2.5 mm cystic nodule filled with colloid was identified in the left thyroid lobe. Right thyroid lobe was not visualized. Subsequently, patient underwent completion thyroidectomy. On histological examination, it was reported as hyperplastic cellular nodule without any evidence of capsular or vascular invasion. The patient postoperative course was uneventful. Patient was put on high dose thyroxine therapy to suppress TSH stimulation which relieved pain to some extent. Patient was discharged with the advice of I-131 ablation therapy for further management. Unfortunately, the patient did not turn up for further treatment. We tried to establish the contact with the patient and patient was doing fine but refused to come for further management.


The patient, a 65-year-old woman, 149 cm in height, 47 kg weight, gravida 4, and para 2, was urgently referred to our hospital for invasive treatment of endometrial cancer following LSC treatment for POP. Full informed consent was obtained from the patient. This study was conducted in accordance with the Declaration of Helsinki (as revised in Fortaleza 2013) and approved by the IRB of Kagoshima University (approval no. 190230).
The patient had visited a gynecological clinic complaining of abdominal discomfort 3 months before being admitted to our hospital. A vaginal pessary for uterine prolapse had been inserted 7 years earlier, which was severely adhered to the vaginal wall and could not be removed. At the time, endometrial cytology was negative, and she was introduced to a urologic clinic for POP surgery. A supracervical hysterectomy, LSC, and perineoplasty were performed. During the operation, the mesh was sutured to the bladder neck with five stitches, and the cervical stump was sutured with three stitches. The other side of the mesh was fixed to the front of the sacrum with two needles and covered with the peritoneum. Pathological findings of a resected uterine specimen revealed endometrioid carcinoma, Grade 1, and negative vessel permeation. However, the POP relapsed within 2 months following surgery; the vaginal wall was relaxed, and the cervix had re-prolapsed exceeding vaginal introitus, which was complicated with cystocele Stage III []. At this stage, the patient was referred to our hospital.
Neither distant metastasis nor lymph node swelling was seen using computed tomography. The patient underwent invasive surgery with a simple trachelectomy, bilateral adnexectomy, anterior-posterior colporrhaphy, and vaginal apex suspension and uterosacral ligament suspension and round ligament fixation in laparotomy. Mesh was sutured to the upper cervix and the suture was not frayed; the resected cervix was complicated by cervical elongation up to 9 cm []. The pathology of a resected specimen did not indicate carcinoma, and washing cytology was negative. Finally, Stage 1A was confirmed, and there was no adjuvant therapy. There were no complications, and no evidence of disease relapse has been reported for 6 months.


A 17-year-old male adolescent who presented with a left scrotal mass was admitted to our hospital. He had discovered the scrotal mass 6 years earlier, but did not seek medical treatment. A swollen left scrotum was identified on scrotal examination, and ultrasonography revealed a 4.7-cm × 1.7-cm left scrotal mass filled with cystic fluid. Ultrasonography revealed a scrotal mass with regular morphology, clear borders and thin walls with detailed hyperechoic areas and poor ultrasound penetration of the cyst’s interior ( and ). A preoperative diagnosis of funicular hydrocele was made, and with the patient's full informed consent, he underwent surgery. Intraoperatively, we identified a 5.0-cm left scrotal cyst with intact cyst wall, and the cyst was fused to the testicular appendix. However, the cyst had no attachments to the left scrotum, and the cyst was successfully removed ( and ).
Intraoperatively, we considered a diagnosis of congenital cyst of the distal urogenital tract. The cyst had a thin wall with no septa and no solid component, on histopathology. The final pathologic diagnosis was a simple serous cyst. However, further histologic examination of the cyst revealed mucin-secreting tall columnar cells, which was consistent with a diagnosis of Müllerian cyst ( and ). The patient recovered well and was discharged on the second postoperative day. One month postoperatively, the patient had no further complaints.


A 58-year-old male presented with a 10-year history of painless right-sided trochanteric mass, which had become progressively uncomfortable and prominent over the past 12 months. There was no history of pelvic trauma or inflammatory disease of the pelvis.
On clinical examination, a painless tender mass measuring about 10 cm in its greatest diameter was noted, mobile relative to superficial cutaneous layers but fixed to underlying structures (). The overlying skin was normal with no signs of infection. Paresthesia in the anterior region of the ipsilateral thigh was reported. The range of motion of the hip was not limited, although a limitation due to moderate pain with passive hip adduction was signaled. All laboratory tests, including red and white blood count with differential count, erythrocyte sedimentation rate, C-reactive protein and rheumatoid factor, were normal.
The patient was initially evaluated with pelvis radiographs that showed no significant patterns in both hip joints. MRI demonstrated a neo-formation of almost 11 centimetres long e 7 centimetres thick, isointense on fat-suppressed T2-weighted images, with hypointensity, relative to fat, on T1-weighted images (). This formation was located in the lateral region of the proximal leg and involved the course of the tensor fascia lata muscle that appeared hypothrofic. The hip joint did not show significant alteration. In the inguinal region there were two lymph nodes of increased size but less than one centimetre.
An ultrasound-guided fine needle aspiration biopsy showed brown granulation-like tissue, with histologic presence of hemorrhagic and necrotic tissue associated to histiocytic infiltrate, suggestive of an old hemorrhagic mass, with no evidence of infection or neoplasia. At this point, we decided to proceed with a wide margin excision.


A 44-year-old caucasian man presented with a solid mass in the right gluteal region. The mass had slowly enlarged over the past 5 years, but recently progressively increased in size. Moreover, he complained of numbness in the right back thigh during the previous month. In medical history, he is not affected by major pathologies and does not take pharmacotherapy.
Physical examination revealed a palpable 10×5 cm hard elastic mass in the right gluteal region. The hip range of motion was no affected but marked hypoesthesia was observed in the sciatic nerve region from the posterior aspect of the thigh to the posterior aspect of the leg. A manual muscle test demonstrated that the strength of the biceps muscle was reduced to 3/5. All laboratory tests, including red and white blood count with differential count, erythrocyte sedimentation rate, C-reactive protein and rheumatoid factor, were normal.
X-ray examination revealed no hip of femoral alteration. MRI demonstrated a big mass of almost 9 centimetres long and 7 centimetres thick, extended from the iliac crest to the great trochanter. The gluteus maximum was laterally dislocated and the mass presented a course towards the piriformis muscle. The MRI signal was isointense to muscle on T1W images and hyperintense on T2W images (). The heterogeneity of the content, of probable haemorrhagic origin, needed further investigation. The hip joint and the other adjacent structures showed no alteration of relief. The ultrasound biopsy shown hemorrhagic and necrotic tissue associated to histiocytic infiltrate that could no orientate towards a diagnosis of certainty.
Also, in this case, we decided to proceed with a wide margin excision, with complete resolution of patient’s symptoms in the postoperative stay ().


A 45 years old man presented to our Emergency Department (ED) complaining for diffuse severe myalgia and weakness. Physical examination showed mental confusion, agitation alternated with drowsiness, oligoanuria, diffuse muscle tenderness, hypotension (90/50 mmHg) and signs of dehydration. No other significant findings on physical examination.
No relevant ECG abnormality were recorded.
The blood tests’ results () were as follows: pH 7.20; pCO2 25 mmHg; HCO3 14 mEq/L; creatinine 10.75 mg/dL; potassium 5.6 mEq/L; alanine aminotransferase 1956 U/L; creatine kinase 551820 U/L. There was no significant increase in troponin level.
He admitted to have abused in cocaine and heroin the day before ED admission. He also reported to be on rosuvastatin 10 mg daily since 3 years before due to familial hypercholesterolemia. No other significant features were found in his past medical history.
Renal ultrasound showed a renal cortex thickness slightly reduced.
Muscle pain was managed with morphine infusion (0.1 mg/kg/hour).
We performed a vigorous intravenous fluid replacement by Ringer lactate (500 mL/h) and 5% glucose solution (500 mL/h) alternately, and electrolyte support with calcium gluconate. Furthermore, 8.4% sodium bicarbonate (100 mL, twice) was administered. Due to severe renal failure, the patient was referred to the nephrologist, who decided to perform hemodialysis.
The patient remained oliguric after first hemodialysis treatment, but parameters and clinical conditions improved, despite persistence of severe myalgia (). Serum CK markedly decreased, but remained significantly high.
Ten days later, the patient was still under hemodialysis with renal function and urine output slowly improving ().


A 51-year-old male presented with sudden-onset, right lower quadrant pain, with gradual exacerbation over 1 day. The patient complained of mild nausea, loose stools without hematochezia and a slightly elevated body temperature (38.5°C). No abnormalities were noted in the patient’s medical history. On abdominal examination, the clinician noticed right lower abdominal tenderness and muscle tension, with normal bowel sounds. The laboratory results were as follows: increased white blood cell count (15 × 109/l) and increased neutrophils (12 × 109/l). Because the symptoms and physical examination were suspicious for appendicitis, an abdominal computed tomography (CT) scan was performed. The CT findings revealed a high-density FB inside the cecum (). The mural of the cecum was thickened, with fat stranding. Foci of gas in the peritoneal cavity adjacent to the object was also observed (), suggested that the FB had caused cecum perforation. The FB was sharp-pointed, with a density of 900 HU (). The patient received an emergency operation, and a punctate perforation area was detected on the cecum. The patient was managed successfully with a cecectomy and the removal of the FB, which was confirmed to be a fish bone (30 mm in length) (). The patient received antibiotic therapy and had no complications during the postoperative period. Upon further investigation, the patient reported eating fish 2 days before symptom onset, which supported the operative findings. The patient was discharged 3 days after surgery.


A 57-year-old man visited our hospital with cough and a high-grade fever (> 39°C) for 5 days. He had a history of contact with a COVID-19 patient 12 days prior. COVID-19 infection was confirmed by PCR, and he was admitted to our hospital the next day (day 1). The patient had been on medication for hypertension and hyperlipidemia, and had smoked until 4 years ago. On admission, pulse was 103 beat/minute, temperature 39.5°C, blood pressure 118/62 mmHg, respiratory rate 28/minute, and oxygen saturation 91% on room air. Blood examination revealed fibrin/fibrinogen degradation products (FDP) 10.6 µg/mL (normal, < 10 µg/mL), D-dimer 2.7 µg/mL (normal, < 0.5 µg/mL); platelets 142×109/L; fibrinogen, 10.6 g/L (normal, 20–40 mg/dL); prothrombin time, 12.6 seconds (normal, 11–13 seconds); thrombin-antithrombin complex (TAT) 3.7ng/mL (normal, 3.75 ng/mL); and plasmin-α2-plasmin inhibitor complex (PIC) 5.4 µg/mL (normal, < 0.8 µg/mL). Chest computed tomography showed bilateral ground-glass opacities. After admission, oxygen therapy was started, but pneumonia rapidly worsened. On day 3 after admission, he was transferred to the intensive care unit (ICU), and mechanical ventilation and favipiravir for SARS-CoV-2 were initiated. On day 7, D-dimer rapidly increased, and continuous unfractionated heparin was administrated. (D-dimer 176.8 µg/mL; platelets 213×109/L; fibrinogen, 8.24 g/L; prothrombin time, 14.2 seconds). A time course of clinical data and therapeutic interventions is shown in .
On day 11, however, the patient was diagnosed with ARDS, and ECMO therapy was initiated. We used the right femoral vein (21 Fr cannula) for blood access and the right jugular vein (16.5 Fr cannula) for blood return. The volume of heparin was increased after ECMO began to prevent thrombosis in the circulation. Bleeding was observed at the cannula insertion point at the jugular vein. Platelet counts rapidly decreased after initiation of ECMO, probably because of the ECMO therapy. After initiation of ECMO, TAT and PIC gradually increased. In fact, this patient was diagnosed with DIC during ECMO treatment, based on the DIC score according to ISTH diagnostic guidelines. Platelets were transfused twice, but were not effective against bleeding.
Therefore, we performed VWF multimer analysis and observed decreased HMW-VWFMs after initiation of ECMO (. Hence, the patient was diagnosed with AVWS caused by ECMO. Loss of HMW-VWFMs can be induced by the degree of shear stress in ECMO and inserted pumps. Shear stress depends on the velocity and viscosity of blood and the diameter of vessels. However, we could not reduce the rotation rate of the ECMO pump or cannula size because of the severe respiratory condition. Therefore, cryoprecipitate made from 1,440 mL of fresh frozen plasma (FFP) was administrated to replenish VWF on day 17. Severe bleeding was temporarily stopped, but minor bleeding continued. In addition, oozing at the anus was observed between day 19 and 23. We performed surgical tracheostomy on day 29 due to the long period of intubation in the mouth. Bleeding from tracheal incision continued despite platelet transfusion. The patient received cryoprecipitate made from 1,440 mL of FFP again, and bleeding subsided. ARDS gradually improved, and ECMO was discontinued on day 31. Platelet counts quickly recovered, and D-dimer, PIC, and TAT decreased promptly. In addition, bleeding symptoms completely disappeared after discontinuation of ECMO therapy.
shows the results of VWF multimer analysis, with plasma levels of VWF antigen and ADAMTS13 activity. The level of HMW-VWFMs dramatically decreased during ECMO therapy between day 14 and day 31. Notably, these findings promptly improved after ECMO discontinuation. Plasma levels of VWF antigen were extremely high, probably due to endothelial cell damage caused by COVID-19. On the other hand, plasma levels of ADAMTS13 activity were moderately reduced, to 20–30% of normal.
Plasma levels of VWF antigen were measured by sandwich ELISA using rabbit anti-VWF polyclonal antibodies (Dako Cytomation, Copenhagen, Denmark) . VWF multimers were classified as HMW-VWFMs if they corresponded to bands > 10 in the VWF multimer analysis as shown in . Plasma values of ADAMTS13 activity were measured by chromogenic ADAMTS13-act-ELISA. VWF multimer analysis was performed according to the method of Ruggeri and Zimmerman with some modifications. Values of 100% were defined as the amounts of VWF antigen and ADAMTS13 activity in pooled plasma from normal healthy volunteers. This study was conducted with the approval of the ethics committee of Nara Medical University. Written informed consent was obtained from the patient.


A 13-year-old female came to the Emergency Department (ASST Settelaghi-Fondazione Macchi, Varese, VA, Italy) with a possible dislocation of the right ankle after a fall from her horse.
At clinical examination, an antero-medial tenderness of the foot, local pain and inability to bear weight on right foot were observed.
Laboratory examinations were normal.
A trauma X-ray series was performed, with no evidence of fractures (Figure ).


A 61-year-old Japanese woman developed nausea, vomiting, and diarrhea.
Three months before the first admission, she experienced nasal congestion, aural fullness, and auditory disturbance, and was diagnosed with otitis media. Also, two months before presentation, she experienced cough worsening, and chest radiography revealed a pulmonary infiltrative shadow. Two months before presentation, she was referred to our hospital for the evaluation of pulmonary consolidation with positive myeloperoxidase-ANCA (MPO-ANCA). She was admitted to the previous hospital. She had a body temperature of 38 °C and elevated C-reactive protein level (7.7 mg/dL). Although she was treated with antibiotics, her symptoms, inflammatory markers, and chest infiltrates did not improve. Laboratory examination showed a high titer of MPO-ANCA (Table ), and she was referred to our hospital. After admission, her creatinine level increased with proteinuria, glomerular hematuria, and granular casts, indicating the presence of rapidly progressive glomerulonephritis (RPGN). Granulomatosis with polyangiitis (GPA) was diagnosed on the basis of the presence of otitis media, sinusitis, pulmonary nodule, RPGN, and positive MPO-ANCA. She received pulsed methylprednisolone followed by prednisolone (PSL) 55 mg/d in combination with intravenous CPA at a dose of 500 mg/mo. Her condition significantly improved, and she was discharged 9 d after the second course of intravenous CPA when the PSL dose was 45 mg/d. She developed nausea, vomiting, and diarrhea the day after discharge, and was admitted to our hospital.
She had been diagnosed with Grave’s disease since the age of 40.
The patient had no family history.
On admission, her consciousness was clear, body temperature was 36.3 °C, and body pressure was 106/75 mmHg. Her abdomen was soft and flat, with pain in the left lower quadrant without defense or rebound tenderness.
Laboratory testing indicated hypoalbuminemia and hypogammaglobulinemia, suggesting protein-losing enteropathy that resulted in the leakage of proteins from the gastrointestinal tract (Table ).
Abdominal radiography did not show niveau or free air. Computed tomography (CT) revealed wall thickening of the stomach, small intestine, and colon with contrast enhancement on the lumen side (Figure ). The contrast enhancement of the outer layer of the intestine was poor, indicating edematous change. Upper gastrointestinal endoscopy showed edematous thickening of the stomach and diffusely distributed erosion throughout the descending duodenum (Figure ). Colonoscopy showed generalized edema and depression with erythema, mainly at the end of the ileum. The depression had no ulcerated surface.
Biopsy showed the absence of malignancy and vasculitis, as well as neoplastic changes including hyperplasia. Mild inflammatory cellular infiltration and some granulation tissues and ulcers were present. Cytomegalovirus staining was negative. Because the differential diagnoses included infectious enteritis, GPA-associated gastroenteritis, and drug-induced enteritis, the patient was initially treated with antibiotics including meropenem. However, hypoalbuminemia progressed. Therefore, we replenished albumin and gamma globulin and performed PE. Her condition did not improve, and her serum albumin levels remained approximately 1.5 mg/dL for > 1 mo. This clinical course ruled out the presence of an infectious etiology. Therefore, we decided to augment immunosuppression, and increased the PSL dose to 60 mg/d and added tacrolimus. Nevertheless, the enteritis did not respond to these treatments, which was contradictory to the autoimmune phenomenon, including GPA-associated enteritis. At this point, infectious and vasculitic etiologies were unlikely.


A 61-year-old male patient was admitted to Hunan Cancer Hospital in September 2018 with a 1-month duration of cough and expectoration. The patient had a 40-year smoking history. Bronchoscopy revealed synchronous bilateral lung lesions in the right and left upper lobes (). Biopsies of the lesion in the right lobe identified squamous hyperplasia with severe atypical hyperplasia, but no obvious metastases were detected. Additionally, immunohistochemical (IHC) p40 staining was positive in both lesions (). Subsequently, lobectomy was performed to remove the left upper lobe and right upper lobe lesions in September and December 2018, respectively. Histology from the right lesion showed severe hyperplasia of bronchial squamous epithelium with no infiltration (). For the left lesion, non-keratinizing squamous cell carcinoma with superficial infiltration was identified (). No metastases were found in the regional lymph nodes or the bronchial stump. These results suggested that the bilateral LSCC was stage IA (tumor, node, metastasis: T1aN0M0). The patient received regular follow-up, with no recurrence or metastasis, and follow-up is on-going.
The bilateral primary tumor lesions were then subjected to NGS targeting 425 cancer-relevant genes (GeneseeqPrime; Geneseeq Technology Inc., Nanjing, China), and their genetic alterations were analyzed and compared (). Mutated genes in the right upper lobe tumor were as follows: discoidin domain receptor 2 (DDR2), fibroblast growth factor receptor 3 (FGFR3), nuclear receptor binding SET domain 1 (NSD1), chemokine receptor type 4 (CXCR4), and TP53 (c.341_352delTGCATTCTGGGAinsG), whereas only a TP53 mutation (c.475G>C) was detected in the left upper lobe tumor (). Therefore, the mutation pattern was completely different between these two tumors and consistent with sDPLSCC.


In July 2020, a 63-year-old man developed fatigue, shortness of breath, dry cough and fever to 38°C. A chest radiograph revealed bilateral bronchopneumonia, more prominent on the left. Nasopharyngeal swab for the real-time polymerase chain reaction (RT-PCR) test for SARS-CoV-2 was negative. However, serology was positive for IgM and IgG antibodies against the virus. His erythrocyte sedimentation rate (ESR) was 75 mm/h, C-reactive protein (CRP) was 103.5mg/l, D dimer was 1281.05 ng/mL, lactate dehydrogenase (LDH) was 505 U/L, and ferritin 1004 ng/mL. His complete blood count was within normal limits (5.3×109 leucocytes, with slight predomination of granulocytes (84%)). Other biochemical analyses were unremarkable, except for an elevated glucose level (8 mmol/l). His oxygen saturation (SpO2) was 92%. Due to high clinical suspicion of COVID 19, he was hospitalized and treated with antibiotics and anticoagulant therapy. His history was positive for arterial hypertension, treated appropriately. Metformin was introduced in his therapy, with additional diabetes mellitus type II diagnosis. Ten days after presentation, he was discharged from the hospital with SpO2 96%, improved chest x-ray findings and improved clinical status. His laboratory values returned to normal over the subsequent two weeks. Seven days later, a nasopharyngeal swab was again negative. Viral serology remained positive for IgM and IgG, and he had lymphopenia (9%).
Four weeks later, he developed blurred vision in his right eye. Initially, visual acuity was normal, but within a week it decreased to 20/630 (0.03) on the right and remained normal (1.0) on the left eye. He had a small papillary edema on the right fundus. The other ophthalmological findings were orderly. At that time he complained of a headache, predominantly on the right side. He was directed to our clinic for suspicion of anterior arteritic ischemic optic neuropathy (AAION).
He was hyperopic (+3.5D OD and +2.0D OS), with additional presbyopia correction. He reported that 20 years prior, he suffered from headaches on the left side, resembling cluster headaches. His personal and family histories were unremarkable. On admission to our hospital, PCR for SARS-CoV-2 was repeated and it was negative. IgM and IgG antibodies remained positive.
Examination of his right fundus was notable for partial swelling of the optic nerve head and ganglion cell layer (). There was a relative afferent pupillary defect (RAPD). Other ocular fundus findings were normal bilaterally. His right eye was almost perimetrically blind, with visual field index (VFI) 1% and mean deviation (MD) −31.56dB; the pattern deviation (PSD) was not shown for a severely depressed field, and left eye had also a sensitivity reduction: VFI of 90%, MD −7.23 and PSD 1.86 dB.
Serology tests were negative for Borrelia burgdorferi, human immunodeficiency virus, herpes simplex, varicella zoster, cytomegalovirus, Epstein-Barr virus and hepatitis B and C viruses. QuantiFERON gold test for tuberculosis was negative. Angiotensin-converting enzyme levels were within normal values. Antinuclear antibody (ANA), antineutrophil cytoplasmic antibodies (ANCA) and rheumatoid factor (RF) tests were within normal range, as were antiphospholipid and anticardiolipin antibodies. An aquaporin 4 (AQP4) antibody test was negative. The following were also normal: ESR, CRP, fibrinogen, D-dimer, coagulation times, ferritin and homocysteine. Plain chest and head radiographs revealed no abnormalities. The only suspicious result which arrived in the meantime was reactive result for Venereal Disease Research Laboratory (VDRL) test. We consulted an infectologist, and agreed with him to do Western blot for T. pallidum
He was treated only with pulse methylprednisolone therapy of 1000 mg for five days in the clinic, with gastroprotection and glucose and blood pressure monitoring. On the fifth day of therapy his visual acuity was significantly better (20/63 or 0.3), and visual field showed significant improvement (). The headache stopped during the first days of therapy.
Brain magnetic resonance imaging (MRI) was performed on the seventh day of hospitalization, in three planes with multiaxial reconstruction, before and after the application of paramagnetic contrast. It revealed only microangiopathic and cortical reductive signs, a neuroglial cyst of 5 mm on the right temporal side, and normal appearing orbits and optic nerves. Spinal medulla on cervicothoracal contrast MRI was without significant alterations, with signs of spondyloarthrosis and hyperostosis.
After hospitalization, he was treated with prednisone tapering therapy for two weeks. On follow-up, three weeks later, he brought a Western blot tests for T. pallidum, which we ordered because of previous VDRL test result. The Western blot was negative for all antigenic bands for IgG and IgM. We consulted a neurologist who did not identified any neurological disorders. His right visual acuity was 20/25 (0.8), and disk edema was resolved entirely. The left eye had normal functions. We ordered MOG antibodies, and two weeks later, we received positive result (titer 1:40, indirect immunofluorescence, cut off 1:10).
At his final 3-month follow up, his visual acuity and computed visual field on the right eye were normal (VOD 20/20 or 1.0; ). The visual evoked potentials (3C PVEP) showed slightly prolonged latency of the p100 wave, and OCT suggested slight thinning of optic nerve fibers and ganglion cells, after optic neuritis (). The MOG antibodies titer was 1:20 and SARS-CoV-2 titers for IgM and IgG were 17.32 and 40.02, respectively.


A 52-year-old male patient with subacute TBAD was referred to our clinic for rapid growth of aorta (5 mm in a week) and intractable pain. Procedure planning and device sizing were performed using a three-dimensional vascular imaging workstation (HOROS v3.3.6; The Horos Project, Public License). The diameter of the supra-aortic branches, distance between the branches, and the clock position of each branch was determined (Figure ). Oversizing was planned as 10% for both proximal and distal ends. Therefore, a tapered 34x26x200-mm stent graft (Ankura™ TAA Stent Graft, Lifetech Scientific, Shenzen, China) was selected for the procedure.
The procedure was performed under general anesthesia in the hybrid operating room. Right femoral artery surgical exposure was performed and left percutaneous femoral artery access was obtained. After heparin administration, pigtail catheter was inserted up to the aortic arch for the true-false lumen control. Following the initial angiographic control of the aortic arch, the stent graft was unsheathed up to one strut distal to predicted fenestration area (5 to 6 cm) on the surgical back table. The endograft covering material (expanded polytetrafluoroethylene [ePTFE]) was fenestrated using an 11-sized scalpel and, after the fenestration, the struts were cauterized for the left over the fabric materials (Figure ). Unsheathing procedure, fenestration, and resheathing procedure took only around 5 min. Approximately 1-cm2 s ingle f enestration was made for LSA by giving a special attention to preserve the endograft integrity (Figure ). While introducing the endograft, marker "8" should be on the greater curvature and seen as a line. Being able to read the marker "8" or "0" during fluoroscopy confirmed the malpositioning of the endograft (Figure ). Once the fenestration was ensured to be oriented toward the supra-aortic target vessel, the deployment was performed. The mean arterial blood pressure was lowered to 50 to 60 mmHg during deployment to optimize accuracy. Completion angiography showed the closure of primary tear, patency of LSA, and no type 1A endoleak (Figure ). At one-month follow-up, computed tomography revealed a perfect positioning of the endograft, no endoleak, ongoing thrombosis of false lumen, and patency of LSA (Figure ).


A 25-year-old male originally presented with mid-back pain of 2 months duration. After a T8 thoracic CT-guided biopsy, he was diagnosed/treated for TB spondylitis with bed rest and appropriate antibiotic therapy. However, 1 month following his initial presentation, he developed worsening of his mid-back pain (VAS score 8), chest pain, shortness of breath, and a progressive lower extremity paraparesis (3/5 proximal and 4/5 distal).
The chest X-ray showed a pleural effusion, while the thoracic spine X-ray demonstrated T8 vertebral collapse. Further, the thoracic spine MRI showed a posterior epidural collection extending from T7 to T9 resulting in cord compression. Of interest, lumbar X-rays and lumbar MR scan both demonstrated an additional although smaller, asymptomatic L3 vertebral body lesion [ and ].
The patient’s progressive myelopathy prompted a T8, T9 laminectomy that included a culture/biopsy of the lesion, epidural debridement, and T6-T10 pedicle screw fixation []. Notably, at the T8 level, there was a nonpurulent, soft, grayish-white material found in the center of the T8 vertebral body that appeared more consistent with tumor than tubercular infection.
The pathology showed only inflammatory granulation tissue, and the staining for all organisms, including Mycobacterium tuberculosis (MTB), was negative [].
Postoperatively, the patient’s back pain (VAS score 2) improved, and he showed significant neurological recovery (e.g., 4/5 function proximally, 5/5 distally). However, within 3 days, he developed an increased left-sided pleural effusion (1700 cc). Tissue culture and MTB DNA reverse transcriptase-polymerase chain reaction of the fluid were negative. Four weeks postoperatively, when he returned with dyspnea and cough, the PET-CT scan revealed a metabolically active osteolytic lesion predominantly involving the T8 vertebral body, with extension into the T7, T9, and T11 vertebral bodies, involving the left 7th rib, as well as multiple other sites [].
Biopsy of the left 7th rib and immunohistochemistry studies confirmed that the lesion was an EMS []. At that point, the patient was referred for chemotherapy (e.g., cytarabine + daunorubicin). Now 1 year following the completion of chemotherapy, he remains asymptomatic, and the EMS has not yet recurred.


A 60-years-old woman reported that she noticed pigmentation of the conjunctiva of the left eye beginning about 10 years earlier. She was suspected of having conjunctival melanoma and visited our hospital in 2006. Our initial examination found that she had no history of ocular or systemic diseases. Her decimal best-corrected visual acuity (BCVA) was 1.2, and her intraocular pressure was normal in both eyes. Slit-lamp examinations showed that the conjunctiva of the right eye was normal but diffuse disseminated pigmentation was detected around the cornea of the left eye (). The cornea was clear, the anterior chamber was normal, the lens was transparent, and no abnormalities were observed in fundus of both eyes. She underwent excisional biopsy combined with cryotherapy. Histopathological analysis by frozen section suggested PAM with subepithelial reactive lymphoid hyperplasia, as there was no melanocytic nests or obvious cellular atypia and pigmentation (). She was followed for 7 years after the biopsy and no changes were observed (). She was then followed at the hospital near her home.
Fourteen years after the excisional biopsy, she felt discomfort of the left eye and noted a conjunctival tumor that was growing. She then consulted our hospital in 2020. Our examination showed that her BCVA was the same; however, a black tumor that extended from the fornix and disseminated pigments at the bulbar and palpebral conjunctiva were detected in her left eye (). A conjunctival malignant melanoma was suspected, and she underwent orbital exenteration and skin grafting procedures at the Kyoto Prefectural University of Medicine Hospital. Histopathological analysis revealed that the tumor was a conjunctival malignant melanoma () but no metastasis was found by a general check-up and her postoperative course was good.


A 64-year-old Hispanic male with a past medical history of squamous cell carcinoma of
the lung unknown stage, history of COVID-19 infection 2 months prior in October
2020, bilateral pulmonary embolism on apixaban and recent hospital admission due to
generalized body weakness arrived at the hospital via air ambulance after an out of
hospital cardiac arrest (OHCA).
On the day of admission, which happened to be Christmas day, the patient awoke short
of breath and was given oral lorazepam and oral morphine by his family. Later in the
evening, family found him unresponsive and called 911. The emergency medical
responder found the patient in cardiac arrest with asystole and proceeded to do
cardiopulmonary resuscitation, intubation, and placed an intraosseous line with a
total of epinephrine 4 mg administered. Upon arrival to us at the hospital, he was
in ventricular tachycardia, ACLS protocol was continued, and a central line was
placed. He was given another epinephrine 1mg intravenous, defibrillation 200 joules
twice and was started on norepinephrine drip intravenous. The time to Return of
Spontaneous Circulation (ROSC) was 5 minutes and downtime was unknown.
After ROSC, on physical exam he was hypothermic, intubated, tachycardic with
decreased breath sounds on bases and not withdrawing to painful stimuli. Laboratory
test showed anemia, thrombocytopenia, hyponatremia, hyperkalemia, hyperglycemia,
transaminitis, and a positive SARS-COV-2-PCR (). The chest x-ray showed a large
right pleural effusion with pneumothorax. Lower extremity venous Doppler was
negative for deep vein thrombosis.
The patient did not have advance directives and three family members identified as
decision makers, all of whom identified Spanish as their preferred language. A
primary physician fluent in Spanish discussed code status with the patient’s family.
We explained that the prognosis was poor based on his comorbidities, OHCA and
unknown downtime. We described that if the patient were to have another cardiac
arrest despite current measures, his chances for meaningful survival would be poor.
The family confirmed understanding using the teach-back method yet decided to keep
him full code, stating “Es luchador,” translating to “he is a
fighter.”
Overnight, the patient required escalation to four vasopressors: norepinephrine,
vasopressin, epinephrine, and phenylephrine. He was transfused one unit of red blood
cells and started on dexamethasone 6 mg intravenous daily.
On hospital day 2, his oxygen saturation decreased to 76% on Fio2 of 100% on the
ventilator. The intensivist performed a right-sided thoracostomy for the effusion
and pneumothorax. Patient’s family was updated and stated they would still want the
patient to remain full code and that they were waiting for a miracle to happen.
Later that day, patient showed no improvement and updates were given to the family
who request a video-call to see the patient. The family decided to discontinue
life-prolonging treatments after this, and the patient passed away 2 hours after
decision was made. No palliative care team was involved during this admission.


A 22-year-old male reported to the department of periodontics with a chief complaint of an overgrowth in his gums that was persistent for a week []. The overgrowth was painless, and mild bleeding on provocation was seen. Previous medical history revealed that the patient was a case of CGD. He was 22 years old but looked very young for his age. He had no facial hair growth, and his limbs and extremities did not appear to be that of a 22-year-old male. He was also short for his age []. He was 5 feet tall and weighed about 40 kg. He was not under any form of treatment for the same.
Gingival examination showed a gingival overgrowth in the lingual aspect of the mandibular left canine involving the interdental papilla. It was ovoid and measured about 10 mm × 8 mm in height and width. The soft-tissue overgrowth was firm in consistency, was mildly mobile, was painless, and did not contain any exudate. The probing depth was 2 mm all around and showed no signs of bone defects and did not warrant any radiographic investigation. A differential diagnosis was given as idiopathic gingival enlargement. To arrive at an exact diagnosis, it was decided to excise the overgrowth and send the specimen for a biopsy. Advanced diagnostic tests were not done on the sample due to poor economic status of the patient.
After adequate local anesthesia, the gingival overgrowth was excised using laser []. Indilase™ soft-tissue diode laser was used to excise the tissue overgrowth. Postsurgical analgesics were prescribed. The excised tissue was sent for biopsy [].
Histopathologic examination showed dense inflammatory cell infiltrate consisting of plasma cells, lymphocytes, and scanty neutrophils and eosinophils []. Also seen were proliferating spindloid fibroblasts and myofibroblasts with numerous capillaries. The final diagnosis was concluded as IMT of the gingiva.


An 88-year-old woman with dementia was brought to our emergency department (ED) due to two rapidly growing painful nodules on her right lower leg. Due to her dementia, the medical history was mostly obtained from her son, and the informed consent for this case report was signed by her son. She had poor performance status and was capable of only limited selfcare (Eastern Cooperative Oncology Group Performance Status Scale grade 3). She was confined to the bed or chair most of the time and was normally cared for at a nursing home. According to her son, the nodule on her right anterior lower leg appeared about 3 weeks previously, and she had begun to complain of right lower leg pain. The other tumor was located at the medial aspect of her right lower leg, and it had appeared only 1 week previously. The two nodules had grown rapidly, with increasing pain. She denied experiencing fever, night sweats or weight loss in recent months. She had been seen at a local clinic prior to her ED presentation. At the clinic, antibiotics had been prescribed and a needle aspiration of the larger skin lesions was attempted. No pus or fluid could be aspirated, and despite antibiotic treatment, the lesions were getting larger. Thus, she was transferred to our ED for further evaluation and treatment.
Physical examination revealed two red and violaceous skin lesions over the right lower leg, ~9 and 3 cm in size, respectively. The lesions were erythematous, had ill-defined borders and were tender on palpation. Local heat and swelling of the skin around the lesions were also noted. Laboratory data, including a complete blood count and biochemical studies, revealed no significant findings. Contrast-enhanced computed tomography of the lower limbs revealed an abnormal space-occupying lesion about 9 cm × 7.5 cm × 3.3 cm in size, at the anterior aspect of the right lower leg. It showed heterogeneous density, heterogeneous contrast enhancement, and involvement of the subcutaneous area, superficial fascial planes and muscle layer. Another small non-specific lesion about 3 cm in size was located at the medial aspect of the right lower leg, with similar radiological features (). With a provisional diagnosis of skin tumors with associated cellulitis of the right lower leg, she was subsequently admitted to our institution for further investigations and antibiotic treatment.
After admission, we performed incisional biopsy of the skin tumors () and the specimens were sent for histopathological examination and bacterial/tuberculosis/fungal culture. An antibiotic (oxacillin 2 g every 6 hours) was administered as empiric therapy for cellulitis. Three days later, bacterial culture and tuberculosis acid-fast stain testing provided negative results. A week later, histopathological results indicated a malignant lymphoma. There was diffuse infiltration of large, atypical lymphocytes with centroblast and immunoblast features in the whole dermis without epidermal involvement. Immunohistochemical staining revealed the following results: CK AE1/AE3(−), CD3(−), CD20(+), CD30(−), BCL-2(+), CD10(− to dim, −/+), BCL-6(dim,−/+), MUM1(+), Cyclin-D1(−), Ki-67(85%) and IgM(−) (). The results were compatible with a diffuse large B-cell lymphoma, either primary or secondary. There was no evidence of lymphadenopathy elsewhere. Based on clinical and histopathological findings, a diagnosis of PCDLBCL, LT was made. The case was later referred to a team of oncologists, and her family refused further management due to old age. Therefore, the patient then received hospice palliative care.


This is a 75-year-old man with a past medical history of diabetes mellitus and hypertension. He had a history of 6 months of an intense left flank pain. Physical exam was unremarkable. An abdominal ultrasound described a left renal mass. Computed tomography (CT) showed a 7.3 × 5 cm mass in the descending colon infiltrating the left kidney, iliopsoas muscle and retroperitoneum. No distant metastases were observed ().
Laboratory tests showed increased serum creatinine (2,0 mg/dl) and glycosylated hemoglobin (8,8%) levels. Carcinoembryonic antigen was normal (2.8 ng/ml). A urine culture was positive for Escherichia coli.
Surgery was decided due to high suspicion of a symptomatic locally advanced colorectal neoplasm. Intraoperatively, a tumoral mass was founded, involving the descending colon, left kidney, lateral abdominal wall and retroperitoneum. A left segmental colectomy with primary colonic anastomosis, left partial nephrectomy and retroperitoneum soft tissue resection was performed. Postoperative recovery was uneventful. Patient was discharged on postoperatory Day 6.
Pathology described a big mass of 8.5 × 5.3 × 3.5 cm infiltrating the pericolic adipose tissue and the colonic mucosa. Microscopically, an extensive chronic inflammatory reaction was observed involving the colonic wall, renal tissue and muscle, corresponding mainly to macrophages and Michael–Gutman bodies (), compatible with malakoplakia.
During follow-up, patient reported resolution of symptoms and no major deterioration in renal function was observed.


Fatigue, back pain, and loss of appetite for 1 mo.
A 75-year-old man with liver cancer recurrence after right radical hepatectomy in April 2019 was diagnosed with HCC. After a multidisciplinary discussion, the patient received a combination treatment of lenvatinib and a PD-1 inhibitor (camrelizumab, 200 mg/bottle; Heng Rui Pharmaceuticals Inc., China). Because the patient developed hypotension and rash on the second use of camrelizumab, it was discontinued. Moreover, the patient showed significant side effects of oral administration of lenvatinib, including diarrhea, fatigue, and loss of appetite, the drug was discontinued. In November 2020, the patient was hospitalized for immunotherapy. The regimen was changed to a PD-L1 inhibitor (atezolizumab, 1200 mg/bottle; Genentech, Inc., United States) combined with bevacizumab therapy. The first 1200 mg infusion was administered on November 10, 2020. Ten minutes into the atezolizumab infusion, the patient reported dyspnea, sudden dizziness, and numbness in his feet and was soon unconscious, with hypotension (56/38 mmHg), a heart rate of 85 bpm, temporal temperature of 36.7 °C, respiration rate of 25 breaths per minute, and oxygen saturation of 92%.
The patient has a medical history of hypertension, diabetes, and coronary heart disease for many years, cerebral infarction for more than 4 years without obvious sequelae, and hepatitis B for more than 50 years, for which he was taking “entecavir”. He had an allergy history of PD-1.
There was no family history of malignant tumors.
Abdominal distension, back pain, concave edema of lower limbs, and no jaundice or palpable masses were observed.
Blood analysis revealed high levels of alpha-fetoprotein (66 ng/mL; normal, < 25 ng/mL), carcinoembryonic antigen (4.8 ng/mL; normal, < 5 ng/mL), and ferritin (355.7 ng/mL; normal, female < 150 U/mL, male < 200 U/mL). Routine blood test showed leukopenia (1.9 × 109/L; normal, 4-10 × 109/L) with predominant neutrophils (62.9%) with normal hematocrit and platelet count. Prothrombin and partial thromboplastin times were normal.
Contrast-enhanced computed tomography and magnetic resonance imaging of the chest and abdomen performed in September 2020 showed multiple intrahepatic tumors invading the inferior vena cava and right branch of the portal vein and tumor thrombus formation in the inferior vena cava and left atrium (Figure ).