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The patient was a 55-year-old diabetic male who developed necrotizing fasciitis of the left foot and leg. After the infection was eradicated, a large soft tissue deficit was present. This deficit was reconstructed with an ALT flap (design in ) with a length of 40 cm and a width of 12 cm. The circumference of the thigh at the midpoint of the flap was 35 cm.\nThe tissue lateral and medial to the defect was extensively undermined. The superficial fascia was closed with 2-0 absorbable sutures in simple interrupted fashion. The cephalad and caudal most aspects of the donor site were easily reapproximated with 3-0 absorbable sutures placed in the deep dermal layer followed by staple closure of the epidermis.\nThe remainder of the defect was not suitable for primary closure utilizing this method. A continuous external tissue expansion system (DermaClose, Wound Care Technologies, Chanhassen, MN) was then applied in the shoelace technique with six anchors as described in the product insert at the cranial aspect of the remaining defect. Mechanical creep could be appreciated within minutes, which then allowed for primary closure in this portion of the wound. The expansion system was then removed and reapplied in a caudal direction and the process was repeated. Simultaneously, an additional external tissue expansion system was utilized in a caudal to cranial fashion until the wound was closed with the expanders meeting in the middle of the incision (Figures and ). The entirety of the wound was closed primarily at the initial procedure. A drain was left in place given the extensive undermining.\nThe external tissue expanders were left in place for one week postoperatively to reduce tension on the closure and to provide further expansion. During this time, the patient experienced a sensation of tightness in the thigh but had no other complaints in regard to the donor site. The expanders were then removed at the bedside with only one dose of intravenous morphine required for the comfort of the patient. The appearance of the donor site at one month postoperatively is shown in . The donor site has now been stable in this appearance for a total of four months.
An 8-month-old female patient was brought to the dentist by her mother with a complaint of an ulcerative growth in the hard palate. Her mother had noticed an erupting tooth in the anterior part of the upper gums when the patient was 3 months old. She also noted a small ulcerated swelling in the same region about 3 months ago. The lesional area was impacted with food debris resulting in foul odor. There was bleeding from the lesional site on manipulation. There was no other complaint except for occasional mild rashes on the scalp and trunk. The patient was afebrile, and active with no disturbance during sleep.\nOn examination, there were no significant extra-oral findings. Intra-oral examination revealed an ulcerative growth present in the alveolus of the right maxilla extending from the region of 51 to 55 t and also extending onto hard palate, measuring 1.5 cm × 1.5 cm in size. The ulceration had a raised edge. The crown of 52 was noted within the lesion and was mobile []. The other teeth present in the oral cavity were the erupting 71 and 81. The clinical impression was a slow growing ulcerative lesion of the right maxillary alveolus extending onto the hard palate.\nThe posterior-anterior and lateral skull views showed a cystic lesion present in the right maxillary region, and a radiological diagnosis of a maxillary alveolar cyst was given. There was no lesion extending to the skull vault.\nComputed tomography (CT) scan showed a nearly spherical mass in the right side of the palate measuring 1.37 cm (anteroposteriorly) × 1.44 cm (width) × 0.63 cm (height). Destruction of the adjacent alveolar process of the maxilla and anterior aspect of the hard palate on the right side was noted along with the loss of normal bony crypts of upper teeth. Tooth 52 was pushed out prematurely. Multiple enlarged lymph nodes were observed in the neck bilaterally. No focal lesion was seen in the visualized lung apices.\nBlood investigation showed no significant findings. During biopsy, all the crowns of the developing teeth were seen floating within the lesion. The dental surgeon was unable to separate the lesion from the surrounding normal tissue; hence tissue had to be obtained as piecemeal.\nThe gross specimen consisted of one large and several small soft tissues. All the soft-tissue bits were white to dark brown in color and firm in consistency. The largest tissue measured 1.3 cm × 0.3 cm × 0.6 cm while, the smaller soft-tissues collectively measured 1.0 cm × 0.3 cm × 0.6 cm in size.\nMicroscopically the soft-tissues showed ulcerated hyperplastic parakeratinized stratified squamous epithelium with a moderately collagenous connective tissue stroma that was diffusely infiltrated with pale staining cells resembling histiocytes. The lesional cells had an indistinct eosinophilic cytoplasm and exhibited indentation of nuclei. Varying numbers of eosinophils, lymphocytes and plasma cells were interspersed among the lesional cells []. Abundant hemorrhagic foci were evident. The lesional cells stained positive for S100 and CD1a []. Odontogenic epithelial cells, dentine and enamel matrix were also present. As the lesional cells were positive for CD1a a diagnosis of LCH was confirmed.\nOur patient was referred to the oncology department where she was staged as a multisystem high risk case. Our patient was totally asymptomatic with the multisystem involvement being totally unrecognized leading to delay in parents seeking treatment. The patient was placed under ARM B protocol for LCH consisting of an initial treatment with prednisolone, vinblastine, etoposide and a continuation treatment with oral 6-mercaptopurine followed by pulses of oral prednisolone, vinblastine and etoposide. Despite the treatment plan adopted, the baby succumbed to her disease within 6 months after the initial diagnosis.
A 9-year-old girl has been presented to our hospital by her parents with anogenital pruritus and with lesions detected in this area by her mother. It was learned that she was the only child of the family, and her personal care was provided by her mother. Her personal history and familial history were normal.\nHer mother told that pruritus had begun approximately one month ago, and at that time, there has been only one single lesion in the perianal area, but the number of these lesions had increased over time, and she had experienced itching even during the sleep for the last 3 months. We learned that the child has been taken to another center for a visit with the same findings 15 days before, and there, she has received cryotherapy, but there has been no regression in the complaints and in the lesion size.\nIn the examination, there were skin-colored warts with the sizes of 3 × 3 mm, 2 × 4 mm ve 10 × 14 mm in three different areas of the perianal region, and of 13 × 12 mm in the right groin. In the right side of the perianal region, there was another area of 10 × 16 mm, slightly more protuberant compared to skin level, with marked margins and erythema, which was different from other lesions. on whole body examination, no sign of trauma was seen and inspection of the anogenital region did not reveal a sign of sexual abuse. On the pulpa of the right thumb, there was a white-gray, hyperkeratotic wart that extended under the nail. It was learned that this wart has been formed approximately 1.5 years before, and the patient received cryotherapy for this wart and anogenital lesions in an external center.\nFor the patient who has been evaluated for condylomata acuminata and warts by the department of dermatology and for sexual abuse by the department of forensic medicine, it was stated that the warts detected in the anogenital region could be developed secondarily to the autoinoculation from the contact of hand genitalia. In the interviews and examination, no sign of sexual abuse was obtained. Mother and father had no history of wart.\nAs the patient has undergone cryotherapy in another center, we waited for one month to evaluate the response given to this previous therapy, but we decided to give a therapy due to few regression of the lesions and excessive increase of itching. Due to the failure of previous destructive therapy, surgical excision was considered to be appropriate, and the lesions were excised under anesthesia and sampled for the histopathological examination. We also take biopsy from the protuberant lesion with marked margins and erythema in the right of the perianal region. The results of the histopathological examination of the surgically removed warts were reported as condyloma acuminate, and the histopathological examination revealed LSK for erythematous area biopsy. No additional therapy was considered to be required for the wart detected on the thumb. For the treatment of LSK, topical steroids, which are administered as a standard therapy, were not preferred for this patient due to the underlying disease described as HPV infection-related wart. For the palliation of the episodes of itching, we begun to give hydroxyzine hydrochloride, a sedative antihistaminic, at a dose of 10 mg/day via oral route, and the parents and the child were instructed to protect the hygiene of the anogenital area and to avoid the contact of hand-genitalia.\nDuring the 2-month followup, no additional lesion was observed in the anogenital area, it was seen that itching episodes were gradually decreased and disappeared, and as a result of the histopathological examination, it was revealed that the area with LSK-like erythema in the right side of the perianal area disappeared. It was determined that the size of the wart localized on the right thumb was reduced, but it did not disappear, and that no additional therapy was planned for the treatment of the lesion.\nThe lesions located in the anogenital region and on the thumb were photographed to include in the forensic file, but the parents consented only the use of the photograph of the thumb lesion in this case report ().
A 16-year-old boy fell while playing basketball and sustained a fracture of his left tibia and a periprosthetic fracture of his right forearm. The tibia was managed with a circular frame and the forearm was fixed with intramedullary titanium elastic nails. Full union was achieved for both upper and lower limbs; however, further surgery was required due to metalwork migration.\nPrevious incisions were utilised to approach the fracture (). The broken plate on the ulna was left in situ due to it being encased in bone, but all the screws were removable (). An intramedullary canal was then created using the combination drilling technique, i.e., regularly switching between 2.5 mm and 3.2 mm drill bits.\nIt is important to continuously check with radiographs in two orthogonal planes that the drill is as central as possible ( and ). If the drill starts to diverge from the midline, then a smaller drill is used to create a new pilot hole to redirect the larger drill. The radius shows a significant deformity that required a corrective osteotomy prior to distal canal creation and nail insertion. By performing the corrective osteotomy, the length of drill insertion was shortened, decreasing the risk of breakage and improving the accuracy of canal creation in the distal segment as we were able to insert the drills directly into it. Titanium elastic nails were then inserted and an above elbow cast applied ().\nOver the following 18 months, the titanium elastic nails began to migrate and cause some soft tissue irritation. It was deemed beneficial to retain the metalwork and so the excess material was trimmed and impacted back into the bone. The nail exit holes then had a cap of bone substitute (Cerament, Bonesupport AB, Sweden) applied to help prevent future migration ( and ). The tibia circular frame had a combination of fixation elements including fine wire and hydroxyapatite-coated half-pins. The half-pins were easily removed as they had not bonded to the bone. The wires proved to be more difficult to extract as the bone seemed to have compressed them. The patient’s function had returned to his preinjury state at 1-year follow-up.\nThe final range of motion was observed, with a maximum pronation of 25° and supination of 60° (). According to the patient, this matched his preinjury range of motion and he was satisfied with the overall outcome ().
This was a 46-year old man that was brought to a psychiatric hospital on account of excessive drinking of alcohol, repeated beating of his wife, destruction of household property, attempted burning of their house and threatening his wife with a cutlass, culminating in her running away from the house with their children. This patient is the husband to the patient described as Case 1. The man had started drinking in secondary school and came from a family where virtually all adult males drank heavily. His father had drunk heavily until he became very old, whereas his uncle had died from problems that resulted from severe alcohol intoxication.\nOn account of his drinking, his work performance had deteriorated progressively because he was spending increasingly more time drinking and recovering from the effects of intoxication. He was doing very poorly in his legal career and his income was very low. Consequently, his family had had to rely on his wife's earnings for their support, and overall up-keep. The patient had suffered several episodes of road traffic accidents on account of driving while drunk. He was a known hypertensive patient and was taking antihypertensive medications regularly. He vigorously denied having any health or social problems from his drinking and resented being brought to the hospital for evaluation and treatment. While in the hospital, he attempted to run away a number of times. He blamed his wife for the recurrent physical abuses she had suffered from him, because according to him, she was engaging in extra-marital affairs, a charge the lady had vigorously denied.\nAt the time of presentation, the patient was withdrawn and aloof most times, and always talked to himself whilst alone. He denied having auditory hallucinations but accused his wife and “her male friends” of trying to harm him. He further stated that he still loved his wife and wanted her to come back to the house. The patient was considered to have met criteria for the diagnosis of alcohol dependence syndrome with hallucinosis, pathological jealousy, and erectile dysfunction.
A 52-year-old male patient reported to Department of Oral and Maxillofacial Surgery with a history of ulceroproliferative lesion involving the lower lip for the last 5 months. He had a habit of smoking 15 cigarettes/day for last 12–13 years and had habits of alcohol consumption 2–3 times a week for last 10 years. The lesion extended from left commissure of mouth upto midline of lower lip involving the labial and buccal mucosa till left 1st molar region []. The oral hygiene was a bit compromised and foul odor was present. The ipsilateral Level IB (Submandibular) group and Level IA (Submental) group of lymph nodes were palpable, enlarged, and not fixed to underlying structure. The provisional diagnosis of malignant neoplasm was made which was confirmed by incisional biopsy which was suggestive of moderately differentiated type of squamous cell carcinoma.\nMagnetic resonance imaging and contrasted computed tomography were the preferred radiography to rule out the extent of lesion in soft and hard tissue, respectively. The infiltration was seen deep into the orbicularis muscle and part of buccal mucosa; however, no bony erosions were noted. The patient was informed about his condition, and surgical procedure was explained enumerating the possible risks and complications of it before surgery. Informed consent for the use of photographs relating to the case, for medical, scientific, or educational purposes, was likewise secured. The ethical committee approval was taken from the local institutional ethical committee approval board.\nUnder general anesthesia, the patient was subjected to the left side supraomohyoid neck dissection and full thickness excision of lower lip with safe margins of 1 cm. The defect was reconstructed with Karapandzic flap. The flap consists of a horizontal incision along the lower border of the defect in the mentolabial sulcus and bilateral perioral incision in nasolabial fold []. The neurovascular bundle was preserved to maintain flap viability. When sufficient mobilization has been accomplished, the flaps were medially oriented to allow closure in 3 planes, namely, oral mucosa, orbicularis muscle, and skin maintaining the integrity of oral sphincter [Figures and ]. The esthetic and functional outcomes were satisfactory; however, there is some microstomia, but it has no impact on the function. Six months after surgery and radiation, the patient has no signs of local recurrence or regional metastasis [Figures and ]. The excisional biopsy report was confirmatory for Moderately differentiated squamous cell carcinoma [].
A 57-year-old female with widely metastatic high-grade serous adenocarcinoma of the ovary was referred to our tertiary palliative care clinic by her oncologist for management of severe cancer-related pain. Following her original diagnosis, she underwent a number of multimodal disease-directed therapies, including surgical resection and chemotherapy. She had significant ongoing mixed somatic and neuropathic pain in the left lower back as well as her pelvis, both sites of known metastatic disease. The back pain was noted to be a deep sharp pain without radiation to her extremities, located primarily around the region of her nephrostomy tube and into the abdomen. She would also have sharp lancinating neuropathic pain in her pelvis due to tumor burden with prolonged periods of sitting, walking, or activity.\nInitial treatment consisted of multimodal pharmacologic therapy including short-acting and long-acting opioids with nonopioid and adjuvant neuropathic agents. Despite these therapies, she had progressive pain and was ultimately increased to an opioid combination of morphine sulfate controlled-release (120 mg by mouth every 8 hours) and morphine sulfate immediate-release (30–45 mg by mouth every 3 hours as needed). Early changes yielded acceptable analgesia. Conversations and medication adjustments continued over the phone, given the distance between the patient's home and our institution. However, over several weeks' time, pain progressed despite further opioid increases. Given concern for escalating pain and poor response to several attempts at altering her opioid therapy, it was advised that she present for a consultation in the palliative medicine clinic. Fortunately, focused discussions with the patient and her family around the patient's goals of care were addressed with her palliative medicine team at every meeting. She was very clear that her top priority in her ongoing medical care was to achieve acceptable analgesia. During the interview in the clinic, the patient noted significant pain with generalized discomfort, a sense of restlessness, and new muscle fasciculations. Her husband accompanied her and noted intermittent confusion. Her physical exam featured delirium (Confusion Assessment Method positive) with diffuse myoclonus.\nOut of concern for opioid-induced neurotoxicity (OIN), the patient was admitted to the hospital for analgesic control and treatment of what was felt to be a toxic encephalopathy. Despite the known shared metabolic pathway (phase II metabolism) between morphine and hydromorphone, the patient's opioids were rotated from oral morphine to parenteral hydromorphone as the clinical picture continued to unfold. Further workup revealed an unremarkable head CT and EEG. Clinical evaluations and laboratory assessments suggested that, in the setting of her declining physical and renal function (creatinine 1.0 mg/dL with prior baseline 0.6 mg/dL and estimated glomerular filtration rate (eGFR) 55 ml/min/BSA with prior baseline >60 ml/min/BSA), the delirium could be the result of an accumulation of active neurotoxic morphine metabolites (morphine-3-glucuronide). In addition, she was dehydrated, constipated, and had not slept in several days. Unfortunately, her delirium persisted despite correction of metabolic derangements, hydration, and an aggressive bowel regimen.\nUrology was consulted, and a nuclear medicine dimercaptosuccinic acid (DMSA) scan revealed minimal function of the left kidney, despite the presence of a nephrostomy tube. Subsequent vascular radiological investigations revealed tumor-induced thrombosis with surmised infarction of her left kidney. It was concluded that the altered renal function due to infarction likely resulted in an inability to adequately excrete the morphine, and the accumulation of polar morphine metabolites resulted in OIN. As exemplified in this case, even with opioid rotation, centrally active metabolites of hydromorphone (hydromorphone-3-glucuronide) theoretically exist, given that it follows a similar phase II metabolic glucuronidation pathway as morphine, although the relative potency and effect of such hydromorphone metabolites are thought to be significantly less than morphine. The patient continued to have signs of OIN and was eventually transitioned to parenteral fentanyl, given that fentanyl is a unique medication that is largely hepatically metabolized into inactive metabolites. Despite common lore that fentanyl and methadone are agents that do no produce active metabolites and therefore do not elicit OIN, case reports of synthetic opioids eliciting OIN do exist in the literature [, ]. Unfortunately, despite aggressive supportive measures and conversion of parenteral hydromorphone to parenteral fentanyl without reduction for cross-tolerance, she continued to have persistent pain with minimal resolution of her systemic neurotoxicity.\nGiven the concern for poorly controlled cancer-associated pain, the palliative care and pain medicine teams collaborated from the initial visit with concerning symptoms of OIN. At our institution, the palliative clinic shares space within the pain clinic, and the collaboration required a simple conversation followed by same day pain consultation. Initially, the shared thought was moving directly to intrathecal (IT) TDD therapy, as this had been discussed over numerous palliative care visits as a potential option with her physicians and was in line with her goals to achieve enhanced pain control with limited side effects. However, given the underlying metastatic disease process and concurrent cancer treatments, she had a metabolic coagulopathy due to nutritional and micronutrient deficits that required correction with vitamin k to assist in the reversal of her international normalized ratio (INR), thus allowing for any type of procedural intervention.\nUltimately, given the persistent multifactorial delirium and rapidly changing situation, the pain medicine and palliative care teams convened a family conference with the husband, the patients' health-care power of attorney, to again address the current situation and make recommendations that seemed consistent with the patient's predetermined goals. Given the rapid progression of her symptoms despite aggressive attempts to correct potentially remediable factors, the shared medical decision-making discussion included moving forward with TDD, given that this therapy was thought to be the best chance for meeting the patients' goals for comfort and hope to have meaningful interaction with family by reducing the burden of systemic side effects from oral or parenteral opioid therapy. The patient's advanced directive was clear that she appointed her husband to make decisions on her behalf if she was ever in a situation where she was unable to provide consent. Informed consent was obtained through her appointed surrogate decision maker and designated health-care power of attorney.\nAfter correction of her metabolic coagulopathy yielding an INR <1.2, she proceeded with placement of an intrathecal TDD system (Medtronic SynchroMed™ II 40 mL pump and Ascenda catheter). The pump reservoir was placed in her right lower abdominal quadrant, and the catheter tip was placed at T10. Her TDD system was efficiently titrated to achieve acceptable levels of analgesia using a combination of opioid (hydromorphone 2 mg/mL) and local anesthetic (bupivacaine 10 mg/mL). The initial postoperative settings were 0.5 mg/day of hydromorphone in a simple continuous mode without bolus dosing (2.5 mg/day of bupivacaine). Within 24 hours of TDD placement, the patient experienced near complete resolution of her toxic encephalopathy and was able to engage in meaningful conversation with her family and health-care teams. There were no signs or symptoms of opioid withdrawal. In the early postoperative phase, she required additional dosing of oral hydromorphone, but was responsive to 2 mg orally every 3 hours as needed for breakthrough postsurgical pain totaling three to four doses per day. Given the robust response to TDD therapy, the patient was able to participate in goals of care discussions with her interdisciplinary palliative care team and elected to discharge closer to home under the provision of hospice services postoperative day (POD) two. Prior to leaving the hospital, the TDD rate was increased to 0.8 mg/day of hydromorphone, and she was provided with a personal therapy manager (PTM) allowing 0.1 mg/dose every six hours totaling four doses per day. The dose remained stable for seven days. As her oncologic disease continued to evolve, her TDD system was adjusted by her hospice provider to accommodate her daily use of oral opioid therapy. The pump was again adjusted on postoperative day nine, yielding a basal rate of 1.4 mg/day of hydromorphone with four PTM boluses of 0.15 mg/dose of hydromorphone every six hours. This dose remained for POD 9–13, and she passed away peacefully with acceptable analgesia at home with her family under the auspices of hospice two weeks after TDD implantation.
A 72-year-old man was admitted to a local hospital with complaints of general fatigue and body weight loss. A computed tomography (CT) scan of his chest was performed and showed lymphadenopathy in his whole body, including axillar, hilar, mediastinal, and retroperitoneal regions (). The patient was suspected of having malignant lymphoma and underwent excisional biopsy in his right groin. One week after the biopsy, he suddenly presented with a complaint of acute abdominal pain. An emergency contrast enhanced CT scan was performed and revealed a massive retroperitoneal hematoma. Consequently, he was transferred to our hospital for the suspicion of ruptured abdominal aortic aneurysm.\nUpon arrival, he was drowsy, pale, and complained of progressively worsening abdominal pain. His blood pressure was 90/50 mmHg, the pulse rate was 110 beats/min, the hemoglobin was 10.0 g/dL, and the oxygen saturation was 99%. He had no past history of hypertension. A contrast enhanced CT scan was performed repeatedly and revealed a massive retroperitoneal hematoma with active extravasation arising from the infrarenal abdominal aorta coexisting with an extensive retroperitoneal mass surrounding the aorta (). The aorta appeared to be almost of normal caliber, with slight expansion. Radiologic interpretations by a specialist pointed out the density difference between the hematoma and the periaortic soft tissue in the retroperitoneal lesion. As a result, we suspected abdominal aortic rupture caused by invasion by the malignant lymphoma and decided to perform an emergency operation. Considering the risk of tight adhesion between the aorta and the lymphoid tissues, conventional open repair could have been technically difficult. Therefore, we chose to perform endovascular aneurysm repair (EVAR) as an alternative.\nAn emergency EVAR was performed under local anesthesia with mild sedation. Initial angiography confirmed an active extravasation arising from the infrarenal abdominal aorta (). An AFX® endoprosthesis (Endologix, Irvine, CA, USA) was placed below the origin of the bilateral renal arteries covering the ruptured aortic segment using standard techniques. In order to avoid type IA endoleak, another extension cuff was placed in the proximal region. Completion angiography showed the endoprosthesis was in a good position and no evidence of endoleak was observed ().\nThe patient’s postoperative course was uneventful and postoperative CT scan demonstrated complete sealing of the rupture point with no evidence of endoleak (). Five days after the treatment, histological examination of the previous biopsy confirmed the diagnosis of mantle cell lymphoma (MCL), which is one of several subtypes of B-cell non-Hodgkin lymphoma. Afterwards, the patient was transferred to a specialized hospital for the purpose of receiving chemotherapy for MCL.
A 35-year-old male patient, with persistent AF refractory to the pharmacologic treatment underwent RFA in our hospital. As a routine practice in our centre, the patient was assessed pre-procedurally by transesophageal echocardiography (TEE) on the day of procedure to delineate anatomy of pulmonary veins and left atrium (LA) and to exclude LA appendage thrombus. During RFA procedure all four PV were individually isolated under guidance of intracardiac echocardiography. RF energy was delivered using a conventional 4 mm and 8 mm tip ablation catheters and a power setting of 30 W, 50 °C around a circular decapolar catheter located at the pulmonary veins ostia. Application of the RF was immediately interrupted when microbubbles were detected by the intracardiac echocardiography (ICE). After a curative ablation therapy patient was kept on aspirin to prevent pulmonary venous or arterial thrombosis and recurrence of AF. Ambulatory follow-up cardiac computed tomography (CT) angiogram was done 3 months following RFA and showed approximately 60% left superior PVS but no intervention was performed because the patient was asymptomatic and he was kept on oral anticoagulation ().\nTwo months later, the patient developed cough associated with hemoptysis, mild intermittent fever, mild left-sided pleuritic chest pain and shortness of breath on exertion. He visited the primary health centre close to his residency where chest radiography was done and showed left upper lobe poorly marginated opacities and minimal left pleural effusion. Further laboratory studies were done to rule out pulmonary tuberculosis (TB) which were within normal limits. A definitive diagnosis was not made and the patient was given broad spectrum antibiotics and referred to pulmonology clinic in our hospital for further investigation.\nPatient presented to our hospital 6 months after ablation without improvement in the symptoms. Non-enhanced CT was done and showed increased parenchymal attenuation and multiple peripheral patchy consolidations in the apico-posterior and anterior segments of left upper lobe associated with minimal left pleural effusion (). Cryptogenic organizing pneumonia (COP), chronic eosinophilic pneumonia (CEP), fungal infection (pulmonary aspergillosis), lung cancer and primary pulmonary lymphoma were suggested as differential diagnosis and ultrasound-guided aspiration of the left-sided pleural effusion was carried out and the obtained specimen was fluid of serous nature. Besides that, lung biopsy was also performed and the histopathologic examination revealed surprisingly intimal hyperplasia associated with multifocal haemorrhagic infarction due to PVO and hypertensive pulmonary arteriopathy. After that contrast-enhanced CT scan was performed with 3D reconstruction and manifested clearly an occluded left superior pulmonary vein (LSPV) (). Occlusion was confirmed by conventional angiography. Ventilation/perfusion scan demonstrated absent perfusion of the involved lung parenchyma and left upper lobectomy was warranted.
A 45-year-old, gravida zero para zero, female presented with a one-week history of a fluctuant mass and erythema in the right superior breast. She had a history of seat belt injury to the right breast seven years prior, and had felt stable masses in the breast for two years prior to presentation. After admission to the hospital, intravenous antibiotic therapy was initiated for symptoms of infection. No family history of breast cancer was noted at that time. The work-up for presumed mastitis began with a bilateral diagnostic mammogram. The provided patient history included a possible diagnosis of cellulitis with imaging to rule out an abscess of the right breast. The ordering physician also emphasized the history of seat belt injury. The admission diagnostic mammogram revealed heterogeneously dense breasts, as well as the presence of fat necrosis in the upper outer quadrant of the right breast at the 12 o’clock position (Figure ).\nNo significant masses, calcifications, or abnormalities were noted in the left breast at that time. Ultrasound of the right breast demonstrated edema with no evidence of malignancy. The patient was diagnosed with cellulitis of the right breast and discharged with antibiotics.\nTwo weeks later, the same patient returned with exacerbated erythema, hardness, and tenderness in the right breast. In addition, she also noted a new lump in her left breast which she had not noticed before and mentioned this for the first time to the radiologist while ultrasound is being performed on the right side. The right breast showed redness, induration, and tenderness in the upper outer quadrant. Subsequent diagnostic ultrasound of the left breast revealed an irregularly shaped hypoechoic mass with microlobulated margins. The mass measured 21 x 18 x 14 mm and was located at the 3 o’clock position, 3 cm from the nipple (Figure ).\nUltrasonography of the right breast revealed only fat necrosis and edema consistent with the patient history. Overall, the imaging was given a BI-RADS assessment of 4C, which is a moderate concern for malignancy.\nUltrasound-guided biopsy of the left breast revealed invasive ductal carcinoma, a moderately differentiated nature, and a grade of two with components of ductal carcinoma in situ. Follow-up mammography was performed, showing proper placement of a marker in the clinically observable mass (Figure ).\nThe patient's care was transferred to an outside facility following a definitive diagnosis. The patient ultimately received bilateral mastectomies with sentinel lymph node dissections and adjunctive chemotherapy.
A 13-year-old boy reported to the Department of Pedodontics and Preventive Dentistry with a history of fall from cycle 2 days back. The child complained of sensitivity in the upper anterior teeth. The medical history of the child was found to be insignificant. Proper immunization schedule was followed for the child. Clinical and radiographic examination revealed Ellis class III fracture (involvement of enamel and dentin compromising the pulp) of the maxillary left central incisor []. No significant hard or soft tissue injury other than tooth fracture was observed. An intraoral periapical radiograph was taken, which showed the coronal fracture with no root fracture or any other periapical changes []. The child was carrying the broken tooth fragment that was confirming adequately to the fractured left central incisor []. The tooth fragment was stored in water and did not show any significant change in color.\nAn immediate endodontic intervention followed by bonding of the fractured segment using the acid etch technique was decided. Single-visit endodontics was performed for the fractured central incisor. An access cavity was prepared and pulp extripation was performed with the help of barbed broaches. After working length determination, biomechanical preparation was carried out with the help of K-files using the crown down technique. Copious irrigation of the root canal was intermittently done with sodium hypochlorite and normal saline. The canal was dried with absorbent point and was obturated with Gutta percha points and Zinc oxide eugenol sealer using the lateral condensation technique []. Now, the pulp chamber was partially filled with restorative Glass Ionomer Cement. Then, the tooth fragment and the remaining tooth structure was prepared for bonding. The tooth fragment was disinfected with sodium hypochlorite solution and then rinsed properly with water. An enamel bevel was prepared all around the remaining tooth structure as well as the fractured margin of the segment and the fragment was reapproximated to check its fit []. An additional internal dentinal groove was also prepared within the dentine of the fractured fragment part, which approximated the access cavity prepared for endodontic therapy of the remaining tooth structure []. Acid etching of the access cavity and the approximating surfaces of the two segments were carried out for 20 s with 37% orthophosphoric acid []. Bonding agent (Ivoclar Vivadent Inc., Amherst, NY, USA) was subsequently applied and light cured for 10 s. The access cavity was filled with composite resin in small increments and light cured for 40 s for each increment. Then, flowable composite ((Ivoclar Vivadent Inc, USA) was applied into the dentinal grove and on the approximating surfaces of the fragment and the remaining tooth. Both the fragments were reapproximated and light cured for 40 s each from the buccal and lingual aspects of the tooth. Flowable composite was applied over the bevel all around the tooth and was light cured appropriately. Finishing and polishing of the tooth was done and the patient was kept on recall []. The patient came for recall visit at 3 months, 6 months and 18 months and the tooth was found to be intact and functional inside the oral cavity in all the three visits.
The patient was a 55-year-old male entrepreneur, who had been experiencing pain of moderate intensity in the mid third of his left thigh for approximately 6 days. He sought emergency care at a hospital in response to a sudden increase in the intensity of the pain combined with swelling at the site of pain. He had a history of smoking equating to approximately 37 pack years. He did not have any other comorbidities. On physical examination he was slightly pale, with tachycardia (120 bpm) and blood pressure at 100 x 70 mmHg. Physical examination by segments was unremarkable for the head and neck, thorax, and abdomen. Vascular examination of the right lower limb found normal auscultation and visual inspection results, with all pulses present and normal. The left lower limb was well-perfused, but there was ecchymosis and a pulsatile swelling between the mid and distal thirds of the thigh, in the anteromedial region ( a). Inspection of the left foot also revealed signs of distal microembolization ( b) and popliteal and distal pulses were absent.\nUltrasonography of the left thigh showed an aneurysmal dilatation of the SFA measuring 5.8 x 5.3 cm and with associated mural thrombi and perivascular accumulations compatible with a ruptured aneurysm ( ). Since angiotomography was not available at the service providing care, the decision was taken to perform emergency surgical treatment.\nDuring the procedure, extensive hematoma was observed involving subcutaneous and muscle tissues in the anteromedial region of the thigh. Once this had been removed, the ruptured SFA aneurysm could be seen ( 3b). There were no obvious signs of active infection. Proximal and distal ligatures were performed and then the aneurysm was resected and samples collected for anatomopathological and microbiological analyses. Revascularization of the limb was then accomplished by interposition of the contralateral great saphenous vein in reverse, with end-to-side anastomosis – taking into consideration the diameter of the femoral artery and the significant destruction of its walls, as illustrated in c. The contralateral saphenous vein was used both because of the probability of associated damage to deep veins in the limb involved in rupture and because of the greater likelihood of injury during dissection, due to anatomic distortions. There were no intercurrent conditions during the procedure\nThe patient was prescribed prolonged, wide-spectrum antibiotic therapy until the results of the microbiological culture of the aneurysmal fragment were available, showing no evidence of growth of microorganisms. The anatomopathological analysis found true aneurysmal walls, with no specific abnormalities. Supplementary imaging exams did not identify any additional aneurysms or any evidence of valve vegetations suggestive of endocarditis.\nAt 1-month follow-up, the patient had palpable distal pulses and was free from pain or other problems.
A fifty-one-year old Caucasian male was admitted to our hospital with sudden onset confusion, dysarthria, and a unilateral facial palsy.\nThe patient was in his usual health prior to admission. His medical history consisted of hypothyroidism, peptic ulcer disease, and previous alcoholism complicated by alcoholic polyneuropathy. Home nursing services noticed an acute onset dysarthria and a unilateral facial nerve paresis so they contacted the emergency medical services. Due to the acute deterioration of his speech, he was admitted as a possible candidate for thrombolytic treatment-flown into the hospital by air ambulance.\nUpon admission the patient was fully conscious but was perceived to be somewhat perplexed. He was oriented for neither date nor time. On examination, pupils were equal and reactive with corneal reflexes present bilaterally. Both visual fields were intact. Yet, his gaze in the midline was dysconjugate with abduction weakness present bilaterally (). This was manifested clinically with double vision on lateral gaze. No nystagmus was observed. There was no facial asymmetry and on clinical testing no facial nerve paresis was noted. The tongue was not deviated from the midline with no appreciable atrophy or fasciculations. Examination of strength was consistent with a generalized weakness but there were no formal pareses. No involuntary movements were seen. Deep tendon reflexes were normal (2+) with bilateral flexor plantar response. The patient was able to localize tactile and noxious stimuli on both sides without any obvious side-to-side difference. His blood pressure was 170/110 mmHg, his ECG showed a normal sinus rhythm without any PQ segment or QRS complex pathologies, and there was however some T wave inversion in the inferior and lateral leads.\nDue to the acute onset of dysarthria and oculomotor pathologies an urgent cerebral CT was performed revealing hypodense changes in the thalamus bilaterally (). A subsequent CT perfusion scan (CTP) revealed hyperperfusion in the same areas () whereas CT angiography of the posterior circulation vessels did not show any abnormalities.\nA complete blood count was noteworthy for a slightly elevated glucose 7.9 mmol/L (reference range 4.0–6.0 mmol/L) and C-reactive protein 14 mg/L (reference range 0–7 mg/L). The remainder of the blood work-up, which included electrolytes, and kidney- and liver-function tests, was normal. Testing for intoxicants in the blood, which included opiates, benzodiazepines, and ethanol, was negative. A lumbar puncture was performed and analysis of the cerebrospinal fluid (CSF) was normal, showing a CSF protein of 0.45 g/L (reference range 0.0–0.5 g/L), 2 white blood cells per microliter (reference range 0–5 cells per microliter), and CSF glucose of 5 mmol/L. A chest X-ray was performed, revealing no pathologies. A transthoracic echocardiogram was also performed with normal findings.\nMRI imaging of the brain showed bilateral hyperdense lesions in the thalamus on the fluid attenuated inversion recovery (FLAIR) series. These lesions exhibited slight gadolinium (Gd) contrast enhancement as did both mammillary bodies (). The MRI showed no pathologies on the diffusion weighted images and the intracranial vessels were normal as visualized by the time of flight (TOF) technique.\nBoth the clinical presentation and the radiological examinations were consistent with Wernicke's encephalopathy. In addition, caregivers of the patient were contacted and confirmed that he had not been eating normally with increasing bouts of nausea and vomiting over a two- week period. Furthermore they informed us that he had been treated for Wernicke's encephalopathy several years ago, at another hospital. The patient was promptly treated with intravenous thiamine for three days. After starting treatment he showed significant clinical improvement on a daily basis. Vitamin B1 treatment was continued orally and the patient was discharged on the fourth day after admission, clinically completely restituted.
In Case 1, a 79-year-old Korean female patient with Fitzpatrick skin type III visited our institution with a complaint of multiple, variable sized skin lesions with itching sensation on the face and the left hand. The lesions were located on bilateral posterior auricular areas, forehead, right cheek, left temple and left hand, and they could be classified into nodular, superficial, pigmented and morphoeic variants and had gradually increased in size over approximately 10 years (). The patient had previously received surgical excision for BCC on the right posterior auricular area at another hospital 7 years ago. Also 1 year ago, she had received surgical excision for a single BCC lesion on the lower lip. The patient was engaged in agriculture and was constantly exposed to direct sunlight for most of her lifetime. She denied any family history of skin cancer, past history of cryotherapy or radiotherapy, exposure to chemical substances including arsenic, dry ice, and she showed no regional lymphadenopathy. The whole body bone scan for systemic metastases revealed no distant metastasis and punch biopsies depicted findings consistent with Bowenoid actinic keratosis. The lesion in the right posterior auricular area was particularly large in size and magnetic resonance imaging scan showed that it was in direct contact with the parotid gland (). All tumors were completely removed with Mohs micrographic surgery (MMS), except for the lesion in the right posterior auricular area, for which wide excision with partial parotidectomy using a fasciocutaneous advancement flap was employed (), as BCCs located in the periauricular embryonic fusion planes have an increased risk of metastasis []. The pathologic results showed lesions consistent with BCC and some lesions consistent with actinic keratosis. Strands of BCC are directly adhered to the parotid gland, and extend to the parotid tissue located at the left bottom of the photograph (). In cases of recurrence of BCC in pre-auricular or postauricular skin, it is very likely that the lesion has invaded the parotid gland []. To make diagnostic confirm for parotid invasion, the patient was consulted by the specialists of multidisciplinary care system and clinically confirmed as parotid invasion of BCC. After 8 months, the patient complained of skin lesions in bilateral temporal areas and left cheek area. Surgical excision of the lesions was performed to obtain negative margins, and the biopsy results were squamous cell carcinoma in situ and actinic keratosis. During the 18-month follow-up period, there were no complications or recurrences.\nIn Case 2, a 43-year-old Korean woman visited our institution with a complaint of multiple, variable sized skin lesions with itching sensation on the face, scalp, right chest, abdomen and right leg (). The lesions could be classified into nodular, superficial, pigmented and morphoeic variants. She denied any family history of skin cancer, past history of cryotherapy or radiotherapy, exposure to chemical substances including arsenic, dry ice, and she showed no regional lymphadenopathy. All tumors were completely removed with MMS, in which wide excision using a fasciocutaneous advancement flap, split-thickness skin graft, and a local flap were employed. All lesions were completely resected by MMS and wide excision, and they were covered with a fasciocutaneous advancement flap and a split-thickness skin graft. Pathologic evaluation confirmed the diagnosis of BCC. Only the abdominal lesion was classified as Stage II, and other lesions were classified as Stage I.
A 12-year-old girl was referred from the general practitioner clinic with a 1-year history of a right breast lump, initially measuring 1cm in diameter and rapidly growing. Recently, the patient started to experience mild tenderness over the lump, but there were no skin changes or constitutional symptoms. There was no significant family history of similar problems. Clinical examination revealed asymmetrical breasts with obvious enlargement of the right breast. A large 11 cm × 8 cm mass occupied almost the whole breast []. The left breast appeared to be developing normally (Tanner Stage II) as no obvious lesions were detected. There were no other clinical abnormalities.\nUltrasonography assessment revealed a large solid homogeneous 6.2 cm × 2.8 cm × 5.6 cm lesion. Magnetic resonance imaging showed a homogeneous 4 cm × 6 cm mass with microlobulation occupying the entire right breast with increasing enhancement on postcontrast images []. A big feeding vessel originated from the chest wall and was believed to be the internal mammary artery.\nHistopathology examination (HPE) through a core biopsy demonstrated features of fibroadenoma; however, we were unable to exclude a diagnosis of phyllodes tumor, and hence, surgical excision was required for full analysis. Following appropriate preoperative counseling, the patient and family agreed to surgery.\nA superior circumareolar incision was used for the best cosmetic outcome. The tumor was located deep in the subdermal layer with a deep margin anterior to the pectoralis major fascia and was well encapsulated and highly vascularized. Normal breast tissue was found inferior to the mass. The lesion weighed 400 g with a resected size of 12 cm × 9 cm × 5 cm [].\nThe postoperative course was uneventful, and the patient was discharged the day after surgery. On gross HPE, the cut section showed a homogeneous brown cut surface with no areas of hemorrhage or necrosis. The stromal mitotic count was <1 per 10 HPF, with focal areas of usual ductal hyperplasia with myoepithelial hyperplasia. There was no clefting or leaf-like pattern seen to suggest a phyllodes tumor. There was no evidence of metaplastic changes. Hence, the final diagnosis was fibroadenoma.
A 20-year-old asymptomatic female, G1P0, with a history of curettage, is presented to the Department of Gynecology and Obstetrics for termination of pregnancy. Her last menstrual period was 17 weeks and 2 days ago. Abdominal ultrasound revealed a clear gestational sac (GS), fetus with heartbeat and placenta previa. Abdominal ultrasound also showed the GS at a distance from the cavity, with a compressed myometrium between the two of them (). The patient received a MRI examination, showing an enlarged uterus of 13.0 cm ×11.7 cm × 7.9 cm, because of pieces of evidence. The MRI demonstrated a foetus with clear organs (), and compressed the lower uterine segment (). The GS was not connected with the uterine cavity and endometrium, but embedded into the myometrium in the right posterior wall of the uterine. A linear hypointensity of the junction zone was observed between the GS and the uterine cavity on T2weighted image ().\nThis result is likely to be placenta implantation as the myometrium cannot be separated from the placenta. The patient was at risk of the uterine rupture and life- threatening haemorrhage. Emergent management should be performed. Performing uterine artery embolization (UAE) and interventional therapy is inadvisable because of the obstructing myometrium between the cervix and placenta. Employing a surgical exploration of the abdomen was decided that was undertaken under temporary balloon occlusion of the abdominal aorta to reduce the loss of blood. The balloon was placed in the abdominal aorta between the opening of renal artery and iliac artery just before the operation. If the area of the focal damage was heavy, the subtotal hysterectomy or hysterectomy was needed. Otherwise, clinicians can perform excision by laparotomy and hysteroplasty. Given the age of patient, clinicians did their best to perform hysteroplasty instead of hysterectomy. The intramural ectopic GS in the second trimester was successfully excluded without life-threatening haemorrhage (). The patient had an uneventful postoperative course. Her β-hCG titre decreased to 1727 mIU ml–1 on the second day after operation, and then to 440.1 mIU ml–1 on the sixth day. She was discharged 6 days after surgery.
A 7-day-old baby boy diagnosed antenatally with a large intra-abdominal mass was investigated postnataly with ultrasound and magnetic resonance imaging. It showed a large abdominal mass probably arising from the mesentery []. On admission, the baby had gross abdominal distension with dilated veins over the abdomen. Clinically, it was a large lobulated mass occupying the whole abdomen except right lumbar and iliac fossa. The alpha fetoprotein was elevated to 20,000 units and the beta human chorionic gonadotropin was 1.2 units and other routine investigations were normal.\nIntraoperatively, a large tumor was adherent to the under surface of the liver and to the hilum, stretching the hepatoduodenal ligament []. Intraoperatively, the vascular pedicle comprising of portal vein, hepatic artery and the common bile duct got injured. It was repaired with the help of a vascular team. The portal vein was sutured with eight zero prolene. The hepatic artery was stented with 1 mm coronary stent and sutured with interrupted eight zero prolene A choledochoduodenostomy was done. The warm ischemic time was 45 min. The child needed massive transfusion and nearly 350 ml of blood was transfused during the procedure.\nThe child was electively ventilated for 3 days. The liver enzymes were elevated in the immediate postoperative period. Liver protective agent N acetyl cystine at the dose of 100 mg daily was infused for 6 days with supportive measures. The child recovered completely and the liver enzymes decreased and the alpha fetoprotein level dropped to 40 units. The child was discharged on the 12th postoperative day with Ursodeoxycholic acid. At the time of discharge, the serum conjugated bilirubin was 3 mg and indirect bilirubin was 3.4 mg. Review after 6 months showed that the liver functions normalized with serum bilirubin of 1.2 mg with conjugated fraction of 0.5 mg. The serum albumin was normal at 3.6 g.\nThe tumor cut surface showed predominantly solid areas with variegated appearance, focal areas of calcifications with a yellowish nodule. Histologically, the neoplasm was composed of skin with adnexal structures, fat, cartilage, bone, muscle, intestinal tissue, salivary gland tissue, and neural elements. Areas of immature intraepithelial elements were also seen which accounts for more than 3 low power microscopic fields. Liver parenchyma was not seen in sections studied. The impression was that of an immature teratoma Grade III [].
The patient was a 55-year-old male entrepreneur, who had been experiencing pain of moderate intensity in the mid third of his left thigh for approximately 6 days. He sought emergency care at a hospital in response to a sudden increase in the intensity of the pain combined with swelling at the site of pain. He had a history of smoking equating to approximately 37 pack years. He did not have any other comorbidities. On physical examination he was slightly pale, with tachycardia (120 bpm) and blood pressure at 100 x 70 mmHg. Physical examination by segments was unremarkable for the head and neck, thorax, and abdomen. Vascular examination of the right lower limb found normal auscultation and visual inspection results, with all pulses present and normal. The left lower limb was well-perfused, but there was ecchymosis and a pulsatile swelling between the mid and distal thirds of the thigh, in the anteromedial region ( a). Inspection of the left foot also revealed signs of distal microembolization ( b) and popliteal and distal pulses were absent.\nUltrasonography of the left thigh showed an aneurysmal dilatation of the SFA measuring 5.8 x 5.3 cm and with associated mural thrombi and perivascular accumulations compatible with a ruptured aneurysm ( ). Since angiotomography was not available at the service providing care, the decision was taken to perform emergency surgical treatment.\nDuring the procedure, extensive hematoma was observed involving subcutaneous and muscle tissues in the anteromedial region of the thigh. Once this had been removed, the ruptured SFA aneurysm could be seen ( 3b). There were no obvious signs of active infection. Proximal and distal ligatures were performed and then the aneurysm was resected and samples collected for anatomopathological and microbiological analyses. Revascularization of the limb was then accomplished by interposition of the contralateral great saphenous vein in reverse, with end-to-side anastomosis – taking into consideration the diameter of the femoral artery and the significant destruction of its walls, as illustrated in c. The contralateral saphenous vein was used both because of the probability of associated damage to deep veins in the limb involved in rupture and because of the greater likelihood of injury during dissection, due to anatomic distortions. There were no intercurrent conditions during the procedure\nThe patient was prescribed prolonged, wide-spectrum antibiotic therapy until the results of the microbiological culture of the aneurysmal fragment were available, showing no evidence of growth of microorganisms. The anatomopathological analysis found true aneurysmal walls, with no specific abnormalities. Supplementary imaging exams did not identify any additional aneurysms or any evidence of valve vegetations suggestive of endocarditis.\nAt 1-month follow-up, the patient had palpable distal pulses and was free from pain or other problems.
A 16-year-old female was admitted in the internal medicine ward with severe and sudden onset pain of the right femoropubic region radiating down to the knee. Her pain began 3 days prior to admission and was exacerbated with movement or light touch. At the onset of pain the affected region was cold and cyanotic. There was oedema of the entire right lower limb. The patient was afebrile, and her left lower limb demonstrated no pathology. The patient reported a preceding soft tissue injury of the right lower limb ten days prior to admission.\nPreceding the rheumatology consultation, the patient was assessed by internists, neurosurgeons, orthopaedic surgeons, neurologists, vascular surgeons, and psychiatrists. Collectively, they ordered a number of investigations including an MRI of the lumbar spine, MRI and MRA of the thighs and pelvis, radiographs of the affected joints, MRI of the brain, CT of the brain, ultrasound Doppler of lower limb, electromyography, and electroencephalography. None of the above clinical assessments and investigations revealed any pathology indicative of a diagnosis.\nRheumatological assessment revealed severe hyperalgesia in the right lower limb in conjunction with cyanosis and reduced temperature. The left lower limb was well perfused and warm. There were no findings suggestive of arthritis. Upon further questioning, the patient reported having had a similar event two years prior to the current complaint. All investigations and clinical assessments from the previous episode revealed no pathology either and were treated with physiotherapy alone.\nA Tc-99m-MDP bone scintigraphy scan was requested by her rheumatologist. The first and second phases of the scan revealed mildly reduced uptake in the right lower limb as compared to the left. The third phase of the scan revealed increased uptake in the area of the right ankle, indicative of increased bone metabolism. These findings, in conjunction with the history, were suggestive of Complex Regional Pain Syndrome (CRPS).\nThe patient was commenced on intramuscular calcitonin but stopped after a single dose due to side effects including persistent flushing, headaches and nausea. Patient refused further drug therapy. Intensive physiotherapy was initiated with improvement of her symptoms after 2 months. The patient was followed-up monthly for 6 months and the current plan is to review biannually.
The patient is a 54-year-old Caucasian male with a past medical history significant for chronic lower back pain for 15 years and basal cell carcinoma, who initially presented with a left sided inguinal mass associated with mild discomfort (). The patient was diagnosed with an indirect inguinal hernia which was repaired robotically using Progrip mesh at an outside community hospital. Intraoperatively, the surgeon noted what appeared to be a large cord “lipoma,” as well as hematoma that was attributed to attempted reduction, and incarcerated sigmoid colon through the defect. The mass was not sent for pathology and was assumed to be a simple lipoma. At 4.5 months postoperatively, the patient returned with a left sided groin fluid collection ().\nAscribing this to postoperative seroma, a 15 French Blake drain was placed with removal of 1 L of serosanguinous fluid without evidence of infection. This fluid was not sent to pathology. Drain placement was complicated by cellulitis 1 week later which resolved with IV antibiotics. Two weeks following drain placement it was removed as the fluid had resolved. At 7 months postoperatively, the patient again noted recurrent fluid collection in the left inguinal site. A follow up CT scan of the abdomen/pelvis with contrast demonstrated a large left sided retroperitoneal seroma with fluid that extended into the left inguinal canal in addition to a new retroperitoneal fluid collection ( and ()). Additionally, a new right sided retroperitoneal fluid collection was noted well away from the original postoperative bed. Bilateral drains were again placed with evacuation of 1140 mL of hematoma. Cytology of the fluid revealed mononuclear mesothelial, monocytic, and histiocytic cells and culture was negative for microbial organisms.\nThe treating medical team felt that the patient’s symptoms were most consistent with mesh complications, and the patient underwent explantation of the mesh. Intraoperatively, fluid was noted to extend below the mesh into the scrotum. A small portion of the mesh was left behind because it was adherent to the iliac vessels. Follow up CT scan unfortunately showed reaccumulation of bilateral retroperitoneal fluid collections, so repeated attempts were made at drain placement without benefit. Eleven months out from the initial surgery, follow up CT scan revealed a cystic mass-like collection at the site of the retained mesh. Additionally, at this time, a culture from one of the drains showed Klebsiella. Thus, the patient underwent open laparotomy with removal of the residual mesh, and found to have purulent material in the retroperitoneum. After washout and recovery with IV antibiotics, the patient continued to have persistent and recurrent fluid collections and underwent two additional laparotomies to break up what were thought to be loculated fluid collections. Approximately 29 months after the original surgery, the patient again had progressive pain and mass in the left inguinal region and was referred to the University of Colorado. CT scans of the abdomen/pelvis were reviewed by a urologist with experience in sarcomas, and interpreted as being suspicious for a malignant cystic mass (). He underwent a third laparotomy, and intraoperatively, multiple cystic, hemorrhagic masses were removed along with the left testicle which was completely surrounded by the mass ().\nThe pathology was reviewed by expert bone and soft tissue pathologists, and arriving at a diagnosis was challenging. The majority of the resected specimens was dominated by reactive-appearing changes, including fibrosis, hemorrhage, dense hemosiderin deposition, and stromal proliferations. Throughout the masses however there were pockets of neoplastic cells. Some foci demonstrated frankly malignant cells that were compatible with pleomorphic sarcoma (). The following immunohistochemical (IHC) studies were performed: ERG, CD31, CD34, D2-40, calretinin, S100, SOX10, pancytokeratin (AE1/AE3), muscle-specific actin (MSA), smooth muscle actin (SMA), ALK1, STAT6, HMGA2, CD43, CD45, CD3, CD20, CD30, CD1a, and CD23. All of these markers were negative on the malignant cells. The initial histologic differential diagnosis included angiosarcoma, based on the hemorrhage and slightly vascular-like growth pattern, but based on the negative IHC results this was ruled out. Additionally, mesothelioma was considered given the anatomic location and areas of apparent surface growth of the tumor, but was also ruled out using IHC. The remainder of the IHC did not show a definitive line of differentiation. Resected lymph nodes were negative for malignancy. Conferring expert soft tissue pathologists ultimately concurred on a diagnosis of a high grade undifferentiated sarcoma. shows different sections of the mass for comparison.\nIn order to help determine the subtype of sarcoma, whole genome sequencing was performed on the tissue sample using Foundation One Heme/Sarcoma panel. The tumor was microsatellite stable with a low tumor mutational burden (1 Mut/Mb). Additional genomic findings included MDM2 amplification, BCL7A rearrangement, FRS2 amplification, and NCOR2 rearrangement. Considering the MDM2 amplification, clinical appearance, and the originating site of an inguinal hernia, the most likely diagnosis was an atypical, highly cystic, dedifferentiated liposarcoma. The patient was monitored with serial CT scans of the abdomen/pelvis as per National Comprehensive Cancer Network (NCCN) guidelines for resected retroperitoneal sarcomas. He did well until 4 months after surgery, when CT scan again showed local progression of disease. Notably, at no time did the patient develop distant metastases to lungs or bones. He was initiated on gemcitabine/docetaxel chemotherapy for three cycles with best response of stable disease, however eventually developed progression of disease. At that time the patient was enrolled on a clinical trial combining doxorubicin with dual immune checkpoint blockade (NCT04028063).
A 35-year-old male patient, with persistent AF refractory to the pharmacologic treatment underwent RFA in our hospital. As a routine practice in our centre, the patient was assessed pre-procedurally by transesophageal echocardiography (TEE) on the day of procedure to delineate anatomy of pulmonary veins and left atrium (LA) and to exclude LA appendage thrombus. During RFA procedure all four PV were individually isolated under guidance of intracardiac echocardiography. RF energy was delivered using a conventional 4 mm and 8 mm tip ablation catheters and a power setting of 30 W, 50 °C around a circular decapolar catheter located at the pulmonary veins ostia. Application of the RF was immediately interrupted when microbubbles were detected by the intracardiac echocardiography (ICE). After a curative ablation therapy patient was kept on aspirin to prevent pulmonary venous or arterial thrombosis and recurrence of AF. Ambulatory follow-up cardiac computed tomography (CT) angiogram was done 3 months following RFA and showed approximately 60% left superior PVS but no intervention was performed because the patient was asymptomatic and he was kept on oral anticoagulation ().\nTwo months later, the patient developed cough associated with hemoptysis, mild intermittent fever, mild left-sided pleuritic chest pain and shortness of breath on exertion. He visited the primary health centre close to his residency where chest radiography was done and showed left upper lobe poorly marginated opacities and minimal left pleural effusion. Further laboratory studies were done to rule out pulmonary tuberculosis (TB) which were within normal limits. A definitive diagnosis was not made and the patient was given broad spectrum antibiotics and referred to pulmonology clinic in our hospital for further investigation.\nPatient presented to our hospital 6 months after ablation without improvement in the symptoms. Non-enhanced CT was done and showed increased parenchymal attenuation and multiple peripheral patchy consolidations in the apico-posterior and anterior segments of left upper lobe associated with minimal left pleural effusion (). Cryptogenic organizing pneumonia (COP), chronic eosinophilic pneumonia (CEP), fungal infection (pulmonary aspergillosis), lung cancer and primary pulmonary lymphoma were suggested as differential diagnosis and ultrasound-guided aspiration of the left-sided pleural effusion was carried out and the obtained specimen was fluid of serous nature. Besides that, lung biopsy was also performed and the histopathologic examination revealed surprisingly intimal hyperplasia associated with multifocal haemorrhagic infarction due to PVO and hypertensive pulmonary arteriopathy. After that contrast-enhanced CT scan was performed with 3D reconstruction and manifested clearly an occluded left superior pulmonary vein (LSPV) (). Occlusion was confirmed by conventional angiography. Ventilation/perfusion scan demonstrated absent perfusion of the involved lung parenchyma and left upper lobectomy was warranted.
A 25-year-old woman with no previous disease was admitted to an emergency department after being stung in a toe by an unidentified animal when she was walking at night in a place infested with scorpions in an urban area near the local cemetery. A few minutes after the sting, she had important local pain and paresthesia of her entire left leg associated with several episodes of emesis, profuse sweating, mild respiratory distress, hypertension, and tachycardia. She first sought primary medical care, with clinical examination showing blood pressure of 220 × 110 mm Hg and a heart rate of 156 bpm. The electrocardiogram showed sustained ventricular tachycardia with very fast heart rate of approximately 300 bpm associated with a right bundle branch block pattern (). Amiodarone was initiated and the sinus rhythm was restored after some minutes, with the patient then being taken to our hospital. During this initial evaluation, no analgesics or antihypertensive agents were administered.\nUpon hospital admission, moderate respiratory distress was observed with a respiratory rate of 30 incursions per minute and oxygen saturation of 89% in room air associated with bilateral rales compatible with acute pulmonary edema. The patient's heart rate was 110 bpm and her blood pressure was 110 × 70 mm Hg. The electrocardiogram showed sinus tachycardia associated with QTc interval prolongation () and chest radiographs demonstrated bilateral fluffy shadows compatible with pulmonary edema and a normal cardiac area (). Laboratory tests showed hyperglycemia, hypokalemia, important metabolic acidosis with lactate elevation, leukocytosis, and troponin I elevation (). Because of this severe presentation, scorpion antivenom (8 ampoules, Butantan Institute, São Paulo) was administered intravenously about two hours and thirty minutes after the incident and local pain was relieved with lidocaine infiltration.\nDuring the first hour after admission, she presented important hypotension (62 × 23 mm Hg), poor peripheral perfusion, and worsening of respiratory distress. A diagnosis of cardiogenic shock was made at that time and dobutamine was started. After this intervention, the hemodynamic status of the patient improved.\nOn the second day, echocardiography showed important depression of the left ventricular ejection fraction (30%) and severe hypokinesia in wall motion, except for the apical region, which was hyperkinetic. At that time, her clinical condition improved considerably and dobutamine was discontinued.\nOn the third day, the echocardiography was repeated and showed a mild depression of left ventricular ejection fraction (41%) associated with hypokinesia only in the basal segments. She received antiremodeling drugs consisting of an angiotensin-converting enzyme inhibitor (captopril) and a beta blocker (carvedilol).\nOn the sixth day, the patient was discharged and instructed to continue to take the above drugs. One month later, she was evaluated at an outpatient service and was found to be asymptomatic. Echocardiography was repeated and found to have fully normalized, and the two drugs were discontinued.
A 19-month-old male child was brought to our pediatric hematology-oncology outpatient clinic for the management of recurrent anemia of unknown cause. There was a history of hydrops fetalis being detected on the patient's fetal ultrasound examination. The patient weight 2.29 kg at birth and had been delivered at 39 + 4 weeks’ gestational age via a vaginal delivery. As a newborn delivery, the patient had cried weakly and had a pale appearance and was consequently diagnosed with respiratory distress syndrome and anemia. He was treated with a surfactant and underwent 6 packed red blood cell (PRBC) transfusions in the neonatal intensive care unit within the first month of his birth. Subsequently, he continued to receive a PRBC transfusion every 3 to 4 weeks for persistent anemia, until he reached 3 months of age. Fortunately, his hemoglobin levels were sustained >9.0 g/dL from the age of 4 months. However, the patient continued to experience wheezing along with repeated episodes of upper respiratory tract infections. At the age of 19 months, there was a recurrence of anemia and he was transferred to the hematology-oncology outpatient clinic. The father informed us regarding the patient's course of admission in a neonatal intensive care unit for the first few months of his life for the management of persistent anemia of an undetermined cause. Other medical history included 2 operations, one each for inguinal hernia and congenital cataract at the ages of 3 and 7 months, respectively. On eliciting the family history, he also disclosed that an older male sibling to the patient had died of apnea at the age of 5 months.\nOn examination, the patient was severely anemic but anicteric. The patient weighted 7.2 kg, had a 75 cm, and his head circumference was 43.5 cm (all parameters <3rd centile for age). His right eye showed strabismus. The systemic examination revealed no abnormalities including hepatosplenomegaly. The developmental milestones for motor skills and language were found to be delayed for age. The patient's hemoglobin (5.4 g/dL; normal range [NR]: 10.5–14.0 g/dL), hematocrit (18.5%; NR: 32–42%), and the reticulocyte count (0.9%; NR: 0.5–1.5%) levels were severely decreased, whereas the mean corpuscular volume was elevated (105.9 fL; NR: 70–90 fL). The total white blood cell and platelet count were normal. On hemoglobin electrophoresis, hemoglobin F level was found to be elevated (12.1%; NR: <1%), along with an increase in the erythrocyte adenosine deaminase level (60.2 IU/L; NR: 8–19 IU/L). The patient's parvovirus B19 serological test (IgM and IgG) results were negative. A computed tomography (CT) scan of the abdomen was performed to rule out other causes of anemia. CT revealed a herniation of stomach and spleen into the thoracic cavity the posteromedial aspect of left hemidiaphragm, and the patient was therefore diagnosed with Bochdalek hernia (Fig. ). No foci of bleeding and other abnormalities were detected in the CT scan. The bone marrow study revealed a normocellular structure with a very high myeloid-to-erythroid ratio of 18:1 (NR: 1.2:1 to 5:1). The erythropoietic cells were observed to be markedly decreased in number, whereas other cell lines were unaffected. The patient's karyotype was 46XY. An echocardiogram showed an atrial septal defect of 5.58 × 3.77 mm in size. The patient's genetic analysis revealed a heterozygous RPS19 gene mutation, designated as c.380G>A (p.Gly127Glu), leading to a diagnosis of DBA. The parents refused genetic testing of the other family members. A PRBC transfusion was administered as the initial treatment, following which oral prednisone was administered at a dose of 2 mg/kg/day for 4 weeks and then tapered slowly. The patient was continued on maintenance oral prednisone at a dose of 0.3 mg/kg on alternate days and has since been observed to sustain a hemoglobin level of >9.0 g/dL without receiving further transfusions. As an operative repair of the CDH was not performed at the time, a follow-up of the condition was planned on outpatient basis.
A 64-year-old female presented to the emergency department secondary to shortness of breath, cough, and associated fever. She had a past history of chronic obstructive pulmonary disease, left upper lobe cavitary lung lesion, and microcytic anemia. The patient was on daily oral steroids for the last several years due to poorly controlled COPD and had recently been released from the hospital two weeks before for a left lower lobe pneumonia. At the time of her prior admission, she was started on vancomycin and aztreonam for her pneumonia and was ultimately discharged on levofloxacin for ten days.\nDuring her emergency department course, her chest radiograph revealed a worsening left lower lobe infiltrate which was later confirmed on computed tomography of the chest. The patient had worsening hypoxia during her course and was eventually placed on BIPAP therapy, and she was started on intravenous vancomycin, levofloxacin, and fluconazole for a presumed hospital-acquired or fungal pneumonia given her recent hospital admission and cavitary lung lesion. Prior to admission, she had no physical exam findings to suggest a fungal infection. She was admitted to the hospitalist service for further evaluation and management.\nWhile hospitalized, the patient continued antibiotic and antifungal therapy, and on day two, aztreonam was added due to a worsening clinical picture. The following day, the patient underwent consultations from infectious disease, pulmonology, and cardiothoracic surgery due to her worsening clinical status and pneumonia with associated cavitary lung lesion. Following consultations, the patient underwent a fiberoptic flexible bronchoscopy with bronchoalveolar lavage which showed a large mucous plug obstructing the left main bronchus but no associated lesions. Cultures from the bronchoalveolar lavage eventually grew Nonomuraea solani, Candida glabrata, and Candida dubliniensis.\nFollowing a protracted hospital course of nine days, the patient was discharged home with cefpodoxime 400 mg twice a day for ten more days and was instructed to follow up with infectious disease within the next two weeks. However, the patient eventually presented to the emergency department one month later with a left-sided empyema status after wedge resection of the cavitary lesion that grew Corynebacterium amycolatum and Staphylococcus hominis spp. on cultures.
A 65-year-old woman with controlled dyslipidemia and hypertension and a previous history of CABG 9 years previously was referred to our center because of the PSA. She had undergone coronary angiography due to symptoms which were suspected to be of ischemic origin in another hospital and during aortic root injection, the PSA was revealed. Coronary angiography showed that the left anterior descending artery and the right coronary artery were occluded at the proximal part; the left internal mammary on the left anterior descending artery was patent and the right coronary artery was filled retrogradely via the left coronary artery system. There was mild left ventricular systolic dysfunction. In addition, a large saccular aneurysm (25 × 55 mm) was detected in the ascending aorta (). These findings were confirmed by multidetector computed tomographic (MDCT) angiography, which illustrated a large pseudoaneurysm in the tubular part of the ascending aorta (maximum aneurysm diameter and PSA neck were 60 mm and 24 mm, respectively) with a calcified wall and without evidence of leak or pericardial effusion (). The diameters of the ascending aorta, arch, and descending aorta were 27, 20, and 17 mm, respectively, and the arch had 3 patent branches. In transesophageal echocardiography, the distance between the PSA and the aortic valve was 30 mm and the diameter of the oval-shaped orifice of the PSA was 20 × 29 mm. In addition, a thin layered clot was visible in the pseudoaneurysm cavity (). Cardiac surgery consultation was done, but the patient refused open repair owing to the high risk of surgery. At this point, we arrived at the decision to use an ASD occluder device in off-label way so as to seal the PSA orifice. Written informed consent, after explanation, was obtained from the patient.\nIn a hybrid operating room setting, under general anesthesia and with transesophageal echocardiography guidance, after open arteriotomy, a 30-mm Amplatzer-like ASD occluder device (Cardi-O-Fix, Starway Medical Technology, Inc. Beijing, China) was delivered via the right femoral artery using a 14-F sheath and pigtail catheter. To avoid the risk of perforation or rupture of the pseudoaneurysm sac by direct guide wire contact, we entered the pigtail catheter into the PSA cavity, with an approach similar to the insertion of the catheter for the left atrial appendage closure device (). Then, the sheath was introduced over the pigtail catheter through the PSA cavity. Finally, after the device was well-positioned in the orifice, aortic root angiography and transesophageal echocardiography showed no residual endoleak ().\nThe patient’s hospitalization course was eventless. Although dual antiplatelet therapy is not necessary for interventional procedures in the aorta considering the high flow pressure, our patient was low-risk for major bleeding and she was discharged healthy on Aspirin (80 mg/daily), off-label usage of Plavix (75 mg/daily for the first 3 months), Nitrocontin (2.6 mg/BD), metoprolol (25 mg/BD), atorvastatin (20 mg/daily), and ranitidine (150 mg/BD). At 3 months’ follow-up, MDCT angiography was repeated and it demonstrated that the device was in the appropriate position without endoleak (). Once more, we visited the patient 1 year after the procedure and she had been asymptomatic and healthy since hospital discharge. The patient refused our request for repeat CT angiography, which can be deemed a limitation in our follow-up period.
A 70-year-old female with no significant past medical history, Eastern Cooperative Oncology Group performance status 0 and no prior incidence of hypersensitivity reactions, was incidentally found to have a small nodule in the proximal right posterolateral vaginal wall after presenting with post-menopausal bleeding. Subsequent biopsy demonstrated a 9 mm invasive melanoma and the patient underwent wide local excision with confirmed negative margins. 4 months later, she developed a right periclitoral mass. Positron emission tomography/CT scan at that time demonstrated focal uptake in this area but no regional/distant metastases (). Excision demonstrated a large submucosal mass of atypical epithelioid cells with evidence of melanin synthesis, consistent with malignant melanoma. Breslow depth was 9 mm (3 mitoses/mm2) with a positive deep margin, and there was no evidence of lymphatic invasion. Her case was presented at a multidisciplinary tumour board and either additional surgery or radiation therapy was recommended to the patient. Owing to the significant morbidity anticipated with additional surgery, the patient opted for radiation therapy. Given the high risk of both local and regional/distant failure, concurrent chemotherapy was proposed. Owing to the historically poor response rates with standard chemotherapy, an immune pathway targeted agent was considered. This non-standard approach was actually initially proposed by the patient. After being thoroughly explained the current standard of care, in addition to the pros and cons of pursuing concurrent radiation and immunotherapy, the patient chose to proceed with combination immunotherapy and radiation treatment. Several studies have demonstrated that local radiotherapy primes and/or enhances an immune response through cytotoxic T lymphocytes. Concurrent immunotherapy may then further enhance the activity and/or duration of the downstream immune response. Given the historically low efficacy of our current treatment paradigms in this disease, as well as the preclinical/clinical rationale to combine radiation and immunotherapy, a strategy of pursuing a combination of ipilimumab with radiation was felt to be reasonable.\nRadiation was planned with intensity-modulated radiation therapy (IMRT) to the vulva and vagina (no elective nodal radiation to the groin) to an initial dose of 45 Gy (1.8 Gy/fraction over 25 fractions), and was to be followed by an electron boost to a total dose of 63 Gy (1.8 Gy/fraction over 10 fractions) at the site of the positive margin (). A thermoluminescent dosimeter was placed on the vulva at the start of treatment to measure the skin dose and read as 1.78 Gy [95% confidence interval (1.66–1.90)], confirming that the planned dose was accurate on the skin. The patient received her first cycle of ipilimumab (3 mg kg−1) 7 days after the start of radiation, and the second cycle was delivered 3 weeks later when the patient was at a dose of 36 Gy. Around this time (3 weeks post ipilimumab cycle 1), she began to develop non-painful erythema in the vulvar and perianal area, as well as a pruritic, grade 2 cutaneous eruption that morphologically showed distinct erythematous papules that coalesced into thin plaques over the upper arms, chest, back and face/ears (all toxicities were graded using Common Terminology Criteria for Adverse Events version 4.03). She did not experience any fevers or other systemic symptoms. By 48.6 Gy dose (10 days post ipilimumab cycle 2), the patient developed a grade 3 skin reaction () that was characterized as a moist desquamation with significant oedema, erythema and pain in the vaginal/vulvar/perianal region and was restricted to the radiation field (). A timeline of these events is illustrated in . After proper consent, a 4 mm punch biopsy of the affected skin was performed and histopathological examination demonstrated spongiotic and interface dermatitis with a perivascular inflammatory infiltrate consisting of numerous eosinophils, consistent with a fixed drug eruption.\nRadiation and ipilimumab were held given the severity of the moist desquamation, and the patient was referred to a dermatologist for evaluation of the ipilimumab-associated cutaneous eruption. It is noted that the patient had no existing risk factors or hypersensitivities predisposing her to an enhanced skin toxicity. The patient was started on 0.1% topical triamcinolone cream along with a methylprednisone dosepak. Given only mild improvement after 1 week, she was started on prednisone 60 mg daily (tapered over 7 days) with oral diphenhydramine for pruritus as needed. This resulted in significant improvement in her cutaneous eruption and pruritus. She then received a third cycle of ipilimumab (4 weeks after cycle 2) and resumed her periclitoral radiation boost without further issues after a 1-month break from radiation treatment. She went on to receive a fourth cycle of ipilimumab after completion of radiation without any complications.\nAt follow-up 8 months post completion of radiotherapy, she had complete resolution of the in-field toxicity and improvement of her ipilimumab-associated cutaneous eruption (). Clinical examination and positron emission tomography/CT imaging 10 months after completion demonstrated no evidence of disease recurrence. Most recently, at her 15-month follow-up, she remains disease- and symptom-free ().\nAt each follow-up visit, the patient has repeatedly expressed satisfaction with her decision to pursue immunotherapy rather than standard chemotherapy to complement her radiation treatment. She admits the initial side effects and intensity of the treatment course were difficult, although she is very pleased with the outcomes and would make the same decision if asked to do so again.
A 65-year old male patient with early gastric cancer was transferred from Aruba to our institution. He had a 3-year history of black stools and anemia. His past medical history included multiple comorbidities: diabetes, chronic renal failure, alcoholic cirrhosis Child A, complete heart blockade and thrombocytopenia of unknown etiology. An upper endoscopy and biopsy revealed a well-differentiated intestinal type adenocarcinoma in the antrum. Endoscopic ultrasonography showed a hypoechoic, 3.2 cm neoplasm, without muscularis externa infiltration and reactive ganglia (). Endoscopic mucosal resection was chosen due to tumor size, stage and comorbidities of the patient. The tumor was fully resected without complications. At the end of the procedure the anesthesiologist had difficulty with ventilation and abdominal distention was observed (). He had a 128/91 mmHg blood pressure and 70 bpm heart rate. An endoscopic revision was done before finishing the procedure, without identification of any macroscopic perforation. A nasogastric tube was placed and therapeutic strategies to improve abdominal-wall compliance were instituted (changes in ventilation parameters, nasogastric suction, change to a supine position and removal of any strap over the abdomen). A plain abdominal radiography in the operating room showed a massive pneumoperitoneum (). Decision of a nonsurgical management was conducted and the patient was taken to the intensive care unit (ICU) for monitoring. The IAP measured by a trans-bladder catheter was 33 mmHg. Six hours after ending the procedure the patient developed dyspnea and anuria. The diagnosis of an abdominal compartment syndrome was established. Given the worsening status, interventional radiology evaluated the patient. A CT scan confirmed the massive pneumoperitoneum without intraperitoneal extravasation of contrast (a & b). A percutaneous decompression guided by CT scan was performed with a pigtail catheter G14 (c & d). Air was immediately released under pressure. Immediately after the procedure, the patient's symptoms and hemodynamic status improved. Diuresis returned after a few hours. The pigtail catheter was closed the first day after placement and taken out at the third postoperative day. Control CT scan revealed no evidence of pneumoperitoneum. Pathology report confirmed the resected specimen had free malignant cell margins and areas of high-grade and low-grade dysplasia. The patient was discharged from ICU at postoperative day 2 and discharged from hospital at postoperative day 5 without further complications.
Our patient is a 19-year-old girl, with a long surgical history of multiple complicated abdominal surgeries. She was diagnosed with Primary Hyperoxalosis for which she underwent same setting liver and kidney transplant. A few years afterward, she was diagnosed with intestinal type diffuse large B-cell lymphoma. After that, she had an ileocecal mass that obstructs the bowel lumen and required oncological resection with right hemicolectomy and anastomosis which was further complicated by anastomotic leakage, wound dehiscence and intra-abdominal sepsis thus requiring emergency exploratory laparotomy and Hartmann's procedure with end ileostomy. The patient then underwent laparotomy, Hartmann's revision and ileostomy site closure. For the purpose of graft preservation, the patient was on long-term immunosuppressants.\nAfter more than a year from the last surgery, she developed a central abdominal bulge at the site of her laparotomy scar; she had midline incisional hernia. Due to the long history of laparotomies and multiple surgical procedures, a multidisciplinary approach was necessary, and a surgical treatment plan was set.\nAn enhanced CT scan was performed and showed thinning of the anterior abdominal wall muscles with severe atrophy of the left rectus abdominis muscle with rectus diastasis of around 10 cm distance (Figure ).\nThe injection was performed by the interventional radiologist with the patient in supine position under ultrasound guidance (Figure ). Injection sites were marked at the anterior axillary line between the costal margin and anterior superior iliac spine according to the technique described by Smoot et al. The area was prepped and draped in a sterile technique which was followed by application of local anesthesia in the form of 1% lidocaine at the skin of injection sites. Under ultrasound guidance, BTA was injected at the three sites on either side of the abdomen. The patient received a total of 300 units of BTA diluted in 150 mL of 0.9% saline with a concentration of 2 units/mL. Each of the six injection sites received a volume of 25 mL. Each of the three injection sites on either side of abdomen were used to target the external oblique, internal oblique, and transversus abdominis muscles. After the procedure, the patient recovered smoothly and was discharged home the next day to return for surgery after 3 weeks.\nAbove the iliac crest, transversus abdominis at anterior axillary line. Mid abdomen, internal oblique at mid axillary line. Below costal margin, external oblique at anterior axillary line.\nPatient went for surgery 3 weeks after Botox injections. The procedure started by infiltrating normal saline for subcutaneous hydro-dissection followed by removing the old scar at the midline. De-epithelization was continued just beneath the skin to raise it above the adherent bowel underneath until reaching the normal fascia on both sides of the abdomen. Then, the abdominal flap was raised in the subscarpal plane above the fascia. Closure of the defect and plication of the recti were done followed by placement of sized on-lay fully resorbable monofilament mesh (Figure ).\nTo further relax the lateral abdominal muscles, a total of 200 units of BTA diluted in 8 mL of normal saline were infiltrated at the same previously injected sites (Figure ). Drains were placed bilaterally above the mesh. Subcutaneous tissue was approximated with interrupted sutures in two layers. Skin was closed with 4-0 Monocryl in subcuticular fashion (Figure ). Patient had no complications and was followed up for the next 18 months with no recurrence.
A 35-year-old female patient presented to our opd with swelling of the left knee for 6 years. Initially, the swelling was small, which gradually increased in size, covering the entire anterior aspect of the knee.\nAlso, she complained of pain and loss of movements of the knee with inability to walk. There was no history of discharge from the lesion, fever, weight loss, or other joint pains. General physical examination was normal except for pallor. Systemic examination was also normal. On local examination, the swelling was solitary, about 20 × 15 cm in size with the skin over the swelling stretched and glistening. On the fifth day of hospital stay, the swelling burst open and blood tinged pus started pouring out. The pus was sent for culture which showed Staphylococcus aureus which was sensitive to piperacillin and tazobactam which was started immediately. All routine blood investigations were carried out. The patient was noted to have severe anemia and her random blood sugar level was 385 mg%. Her serum calcium and alkaline phosphatase levels were within normal range. X-ray and MRI scan of left knee showed a well-defined T2 hyperintense expansile eccentrically located osteolytic lesion involving the metaphyseal region of the proximal tibia and extending into the subarticular space and multiple T1/T2 hypointense septations were noted within the lesion suggestive of osteoclastoma (). Her hemoglobin was then improved by giving her serial packed red cells transfusions. She was not a known case of diabetes mellitus and her sugar was controlled by putting her on regular insulin subcutaneously as per sliding scale every 8 hours. Chest X-ray and bone scan were done to rule out any metastasis (). The patient was counseled regarding the tumor and prognosis and various treatment options but due to financial constraints the patient was treated with above knee amputation (). Sample from the lesion was sent for histopathological reporting which showed multiple multinucleated giant cells in a background of stromal cells, confirming it to be giant cell tumor. Elastic stockinette was wrapped over the postoperative dressing to prevent edema accumulation in the residual limb. Postoperatively, she was asked to lie supine or prone (to tolerance) to prevent hip flexion contracture. She was continued on sliding scale as mentioned before till the sugar levels returned to normal. Cardiovascular training was initiated as appropriate to patient tolerance in order to improve endurance and functional mobility tolerance. The patient started on bed mobilization and therapeutic exercises. Ambulation was started on the 5th day as tolerated with assistive device.\nDistance of ambulation was progressed to patient's tolerance. Exercises were encouraged when the patient was on bed or sitting in chair. Dressings were done on the 3rd, 6th, and 9th postoperative days. Pain was controlled with adequate intravenous analgesics. Intravenous antibiotics were given for 4 days after which oral antibiotics were continued till suture removal which was done on the 12th postoperative day. There was no discharge or gaping at the stump site. Patient was then discharged on the 14th postoperative day after giving her oral hypoglycemic drugs. The patient returned for followup after 1 month and had no residual symptoms with completely healed stump (). Ambulation activities with a prosthesis began during the 11th week after amputation after meticulous preprosthetic management.
A 2-year-old male patient presented at the clinic with complaints of a swelling on the left side of the neck, which had been present since birth and seemed to have increased in size over the previous 2 - 3 months. Physical examination determined a mass lesion in the left cervical region reaching approximately 6 cm in size, slightly mobile and pressurized, with no changes in skin color or temperature, leading to posture impairment of the neck. In the laboratory findings, in the acute phase reactants there was a slight increase in lymphocytes and a mild level of iron deficiency anemia. Other routine laboratory results were normal. B-mode USG examination was directed to the cervical region. A complex cystic lesion was observed to be located in the left anterior cervical region, extending intrathoracically.\nMRI examination was made to evaluate the extent of the lesion and details of the internal structure. From the MRI examination, the lesion was determined to be located on the left half of the neck in the infra-suprahyoid region, extending into the retropharyngeal cavity, to the posterior cervical triangle and the intrathoracic left superior mediastinum. The internal jugular vein was completely surrounded by the lesion. The multilocular, septated lesion with lobular contours and thick walls was separated from the surrounding tissue with clear borders. In T1-weighted images, the lesion was seen on intermittent signals depending on hemorrhagic and/or proteinous content and on T2-weighted fat suppressed images there was evident hyperintensity. On contrast T1-weighted fat suppressed images there was only contrast of the thick septa and wall (, ). Although neighboring anatomic structures were compressed due to the mass effect of the lesion, there was no significant compression effect. These findings together with the cervicomediastinal location led to an initial diagnosis of cystic hygroma being considered. The cytology result of aspiration from the lesion was consistent with cystic hygroma. As the lesion was widespread, wrapped around vascular structures and of a macrocystic structure, the decision was made for percutaneous treatment. The lesion contents were aspirated percutaneously. In the same session an injection of sclerosing substance (bleomycin) was administered. At the 3-month follow-up examination, the lesion was determined to have nearly completely reduced.
A 79-year-old retired man with a past medical history of atrial fibrillation, cerebrovascular accident, hypertension, hypothyroidism, and myocardial Infarction presented to the hospital with a one-week history of generalized weakness and hyponatremia on routine blood work that had recently been ordered by his primary care physician.\nOn further questioning the patient admitted to having more shortness of breath recently with minimal exertion (walking 2 blocks) but there were no acute changes prior to admission. The patient denied any fevers, jaw pain, localized headache, or visual changes.\nOn physical examination vital signs including temperature were within normal limits with the patient noted to be in rate controlled atrial fibrillation and normal pulmonary examination. Abdominal examination revealed minimal left upper quadrant tenderness. Neurological exam was normal and there was no evidence of temporal tenderness. The examination of peripheral joints was normal.\nHis admission laboratory results revealed a hemoglobin of 12.1 and his sodium was 123 with normal renal and liver function tests.\nGiven the lack of any symptoms at this stage indicating a possible vasculitic or rheumatological cause for his presentation, the workup at this stage concentrated on looking for an underlying cause and correcting his hyponatremia. Serum osmolarity on admission was 269 msom/kg and urine osmolarity was 219 mosm/kg and urine sodium was 50 mEq/L giving the conclusion that the patient was hypotonic and hypovolemic. This was corrected with saline with dextrose.\nA routine chest X-ray on admission revealed a 6 mm nodule and this provoked a CT scan of the thorax which revealed a few more nodules later deemed benign and possible aortic dissection. CT angiogram of the thorax revealed no dissection but possible aortitis of the upper abdominal aorta. Then the patient underwent CT angiogram of abdomen and pelvis which revealed atherosclerosis of the abdominal aorta but no evidence of aortitis.\nGiven that the possibility of abdominal aortitis was previously raised on imaging a sedimentation rate was ordered which was raised (109) and the patient was worked up with ANA, ANCA, serum protein electrophoresis, and RPR but these all proved to be negative. As a result despite the patient having no typical features of GCA it was felt this should be excluded given the lack of any other possible explanation for his raised sedimentation rate. An initial ultrasound of his temporal arteries was ordered which was normal. The patient was reluctant to have any invasive biopsies done and also the need to hold the patient's warfarin for the procedure created additional risk for the patient. As a result MRI brain with GCA protocol was ordered with and without contrast and this showed mural wall thickening of bilateral superficial temporal and superficial occipital arteries indicative of GCA (Figures and ). The patient was commenced on prednisone 60 mg daily and following this introduction the patient felt constitutionally better over the next 72 hours and was subsequently discharged home on the current dose with rheumatological follow-up.
A 68-year-old woman with a previous history of paraplegia after traumatic spinal epidural hemorrhage from a ruptured L3 arteriovenous fistula (AVF) was admitted to our institute with high fever above 38℃ and severe back pain. About 10 days earlier, she suffered a superficial burn to her back and was admitted into the local hospital because of acute renal failure due to rhabdomyolysis. On neurologic examination, the patient was alert but mildly disoriented. Examination of the motor system revealed intact muscle strength in the upper extremity and flaccid paraplegia with areflexia. An examination of the sensory system revealed that all sensory modalities had decreased below the T10 dermatome level. An abdominal computed tomography scan demonstrated a suspicious pyogenic spondylitis of multiple lumbar spines with multiple perivertebral abscess formation and fluid collections around the pyriform muscles and parasacral area. A drainage catheter was inserted by a radiologist and the patient was admitted into the infectious diseases ward for antibiotic therapy. Empirical antibiotic therapy with ampicillin and sulbactam was started. On culture of aspirated fluid, methicillin-sensitive Staphylococcus aureus was identified. Thus, the antibiotic was changed to nafcillin. Drug fever developed after nafcillin was given, so the antibiotic was changed to cefazolin. This antibiotic was maintained until the culture of the draining catheter was identified. On culture of the draining catheter, methicillin-resistant Staphylococcus aureus and Pseudomonas aeruginosa were identified. Therefore, the antibiotic was changed to a combined vancomycin and ciprofloxacin regimen. After 14 days on that regimen, the antibiotic was changed to teicoplanin because the patient developed a skin rash. Despite conservative therapy using the appropriate antibiotics for 3 months and surgical drainage being performed twice, the spondylitis of her lumbar spine was not controlled. We thought that the fluid collection in the dead space between the intra-/inter-muscular area and the scar of the previously embolized AVF was the main source of infection recurrence (). We believed that by filling the dead space, we would be able to control the spondylitis. Therefore, we designed a staged operation.\nFirst, we performed multiple posterior screw fixations from L1 to the sacrum with wide debridement of infected soft tissue and obliteration of dead space using RLDMF (). Second, we performed anterior column reconstruction with an expandable mesh cage via an anterolateral reptroperitoneal approach to correct deformity and bone fusion. After the staged operation and antibiotic therapy, the multiple infectious spondylitis completely resolved without recurrence () and there were no signs of spinal instability or pseudoarthrosis ().
A 29-year-old female was referred to our hospital because of a intermittent occurrence of right flank pain for 1-week. The patient had neither a family nor personal medical history. Aside from the flank pain, the patient had no other urinary symptoms such as hematuria or mucusuria. Biochemistry and hematologic results were within normal limits. A computed tomography scan of the abdomen and pelvis revealed an ovoid cystic mass located just anterior to the dome of the urinary bladder with a partially calcified wall (). Radiologically, adenocarcinoma arising from urachal remnant was suggested. The patient underwent a radical excision of this mass including the attached portion of the urinary bladder and median umbilical ligament. At laparotomy, the mass originated from the dome of the urinary bladder without connection to the bladder cavity. There was no evidence of pseudomyxoma peritonei or tumors of other organs including ovary and appendix. The patient has been followed-up postoperatively with no evidence of recurrence for 11 months.\nGrossly, a multilocular cystic mass with abundant mucoid materials, measuring 5.5×3.5×1 cm, was found, which was connected to a small cuff of urinary bladder wall and a segment of median umbilical ligament (). The mass did not show any papillary or solid lesions and did not communicate with the urinary bladder lumen. Histologically, the cyst was lined by a tall columnar epithelium with cellular crowding and stratification (). The epithelium showed changes ranging from simple benign columnar epithelium to areas of dysplasia. Cytologically, the lining columnar cells had abundant mucin in the cytoplasm and exhibited loss of polarity, nuclear pleomorphism, and hyperchromatism in dysplastic areas, but there were no mitotic figures. No stromal invasion of the tumor cells was observed, although mucin was extravasated into the cyst wall (), and foci of dystrophic calcification were present. The underlying stroma was composed of hyalinized connective tissue. On the basis of the proliferation of atypical mucinous cells and the absence of stromal invasion, the tumor was diagnosed as a mucinous tumor of uncertain malignant potential. Its association with urachal remnant and location just anterior to the dome of urinary bladder confirmed the primary urachal origin of this tumor.
A 76-year-old woman presented to our hospital with a mass occurring on the skin of her right chest wall. She had been diagnosed with right breast cancer (T1N0M0, stage I) 9 years previously and had received breast-conserving surgery, sentinel lymph node biopsy, and adjuvant chemotherapy and radiation therapy for the residual whole right breast at a previous hospital. She then developed pigmented skin on her right breast 6 years after surgery, and this lesion was diagnosed as an angiosarcoma. The patient underwent a breast mastectomy to treat for RAAS. Following this, however, the angiosarcoma on her chest wall recurred three times within 2 years. The angiosarcoma was resected each time, and she received radiation therapy to her chest wall after the third operation. Four years after the first occurrence of RAAS, we observed light pigmentation and a dark red tumor (gross diameter of 5 mm) on her right chest wall (Fig. ). Clinically, recurrence of RAAS was suspected, and recurrence of angiosarcoma was diagnosed by biopsy. We considered that it was necessary to remove the irradiated skin as much as possible in order to cure the RAAS. After extensive resection of the irradiated skin and tumor, new skin collected from her right thigh was grafted to the site (Fig. ). Pathologically, the tumor size was 6 mm and the surgical margin was negative. Histologically, there were many spindle cells and dilated vascular channels. Immunostaining showed that the tumor was CD31-positive and mildly positive for CD34 (Fig. ). Ki-67 index was also high. It was revealed that there is no inconsistency as recurrence of RAAS is pathological. After the operation, the patient was hospitalized for 30 days and did not experience any complications. Although some reports suggest chemotherapy can be used to treat RAAS, we considered that this option would offer little benefit in this case, because the patient was elderly and had a history of cerebral infarction. Indeed, the patient has remained angiosarcoma-free for the last 3 years following our intervention, even without chemotherapy (Fig. ).
A 34-year-old female patient prediagnosed with G6PD deficiency complaining of gingival swelling and bleeding since 9 years and progressive spacing between maxillary and mandibular anterior teeth reported to the outpatient department of Periodontology of the college.\nHistory dates back to the year 2002 when the patient developed some bleeding complications during her pregnancy and was admitted in the hospital. During her stay in the hospital, the patient showed signs of petechiae and ecchymosis on the extremities. The patient also developed oral symptoms like gingival bleeding and palatal ecchymosis. It was then that she was diagnosed with Aplastic anemia/pancytopenia with hypocellular bone marrow and G6PD deficiency. She was also referred to their dental department for consultation regarding her oral condition. Hospitalization further deteriorated her oral hygiene, thus complicating her already compromised oral condition. Her platelet count kept decreasing and at the time of delivery her platelet count was 5000 only. The newborn boy was also diagnosed with G6PD deficiency but was asymptomatic.\nThe patient was then hospitalized for another 6 months after delivery during which her physical condition and her platelet count improved. Since then the patient experienced gingival swelling and bleeding and progressive spacing between her upper and lower anterior teeth. She visited several general dentists but because of her underlying medical problem she was refused treatment by some while some provided her oral prophylaxis on her written consent. She developed some improvement in the oral condition but finally she was referred to the department of Periodontology of the college for expert opinion.\nIntra-oral examination showed signs of gingival inflammation, the changes being more pronounced in the maxillary and mandibular anterior region. The gingiva was dark pink, edematous, and enlarged with rolled and rounded margins with less pronounced scalloping. There was loss of stippling and the gingiva was smooth and shiny. Also, gingival recession and clefting was present on the palatal aspects of maxillary anterior teeth [Figures and ]. There was pus exudation on palpation. Pathological migration was also evident in the same region.\nRadiographically, generalized bone loss was evident which was more pronounced in the maxillary and mandibular anterior region [].\nClinically, deep periodontal pockets were present in the maxillary and mandibular anterior teeth on probing using a periodontal probe with Willams markings.
A 72-year-old man presented with a 1 month history of a painless palpable mass in the right inguinal region. On physical examination, an irreducible tender mass was detected in the right inguinal region. Laboratory data were within normal ranges. A clinical diagnosis was made of a right inguinal hernia. The inguinal US revealed an oval shaped, mixed echogenic mass () with a focal area of slightly increased vascularity on a color Doppler image. A longitudinal scan showed a hyperechoic area on the superficial one-fifth region, a linear structure on the intervening portion, and a mixed echoic area on the deep four-fifth region of the mass (). There was no evidence of a herniated bowel loop or omental fat. Contrast enhanced multi-detector (MD) CT demonstrated an approximately 9.8 × 5.3 cm well-defined elongated, homogeneously low attenuated mass without an enhancing portion attached to the right spermatic cord and extending to the right inguinal subcutaneous region (). The axial image showed the focal fat density area without an enhancing portion, on the anterior peripheral portion of the mass (). There was no radiographic evidence of any regional lymph node involvement or invasion involving a contiguous structure on CT scan. Preoperative diagnosis of benign soft tissue tumor of the spermatic cord was suggested because the imaging appearances were not compatible with inguinal hernia or inguinal hydrocele.\nUpon inguinal exploration for this suspected soft tissue mass, a 10 × 6 cm soft gelatinous mass was discovered which was adherent to the spermatic cord but simultaneously mobile and free from attachments with other soft tissue elements. The spermatic cord coursed through this mass. Upon closer inspection there was no evidence of a hernia or laxity in the posterior wall of the inguinal canal. The patient underwent excision of the mass without hernia repair surgery.\nThe histopathologic findings showed a well-demarcated, myxoid neoplasm comprised of spindle and stellate cells, mixed with mature adipocytes and numerous thin- and thick-walled vessels of various sizes (). Immunohistochemically, the cells of the myxoid areas were positive for CD34 () but not for S-100 protein, desmin, or smooth muscle actin (SMA). The mature adipocytes were focally positive for S-100 protein (). The CD34 and SMA highlighted vascular endothelial cells and perivascular smooth muscle fibers, respectively (). The tumor was negative for desmin (). On the basis of these findings, angiomyxolipoma was diagnosed.
A 49-year-old woman with a history of type 2 diabetes mellitus for a few years presented with chronic abdominal discomfort and nausea for 1 year. She was treated for peptic ulcer disease but did not get better. Three months earlier, she complained of severe abdominal pain, significant weight loss, and chronic intermittent watery diarrhea. Abdominal sonography showed a large epigastric mass, 10×16 cm in size, with multiple hyperechoic nodules at both lobes of the liver and peripancreatic lymphadenopathy, suspected to be metastatic spread to the lymph nodes. Blood test results were within normal limits, and only hyperglycemia was noted. Blood tests for measuring hormone levels were not available in our institute. The patient was scheduled for a computed tomography (CT) scan of the upper abdomen. While she was waiting at the radiology unit, she developed hematemesis and was referred to the internal medicine department for further treatment. She was admitted, and the gastric lavage showed the presence of blood. Her hemoglobin level was 10 mg/dL. Endoscopy showed a large mass, approximately 15 cm in diameter, in the upper gastric body. The mass extended down to the antrum and duodenal bulb with blood oozing (). Next, endoscopic ultrasonography was performed, and a large well-defined isoechoic gastric subepithelial mass more than 13×8 cm in diameter was detected (). The mass arose from the third and fourth layers of the gastric wall. Multiple intra-abdominal and peripancreatic lymphadenopathy were detected, and the lesions varied in size from 8 to 25 mm. Fine needle aspiration was performed on the mass, and the cytology results led to a suspicion of malignancy. The CT scan showed a large soft tissue mass, with multiple liver metastases and intra-abdominal lymphadenopathy (). Finally, the patient underwent surgery. Gross pathology showed tumor involvement of the posterior wall of the stomach, with the tumor forming a large intramural multinodular mass (). Multiple well-circumscribed masses were detected at the pancreatic head and body and at the first part of the duodenum. Multiple liver nodules were also noted. Histopathology showed that the tumor was arranged in solid nests or in an acinar pattern infiltrating from muscularis propria of the stomach through the mucosa and extending to the duodenum. The tumor cells were cuboidal in shape with ample granular eosinophilic cytoplasm. Their nuclei were monotonous and bland, with salt and pepper nuclear chromatin, a typical characteristic of neuroendocrine tumors including positivity for typical characteristic of psammoma bodies (). Immunohistochemical staining for synaptophysin showed diffuse positivity in the cytoplasm (). Only a few tumor cells showed scattered positivity for somatostatin on immunohistochemistry (). A definitive diagnosis of somatostatinoma was made. The patient underwent distal gastrectomy with Billroth I anastomosis and cholecystectomy. However, she developed acute jejunal obstruction 2 weeks later due to intussusception from the jejunal leiomyoma and underwent gastrojejunostomy. She refused any further adjuvant chemotherapy or hormone therapy and received only palliative treatment. She passed away 8 months later because of massive liver metastasis.
An 18-year-old Japanese male patient was urgently transported to our hospital following a traffic accident. The patient was sitting in the passenger seat with the seat belt fastened and had a car-to-car accident. The patient was diagnosed with right kidney injury, right adrenal injury, and abdominal aortic aneurysm and was admitted to the hospital. Intra-abdominal hemorrhage due to renal injury was controlled by endovascular treatment. The abdominal aortic aneurysm was followed without any intervention. Other organs, including the small bowel, did not show any damage on the first computed tomography (CT) examination. The clinical course of the patient was good after these treatments, and food intake was initiated on the tenth hospital day. However, on hospital day 13, the patient vomited bile-like fluid twice. He experienced no abdominal pain, and hematological tests did not reveal any abnormal findings. CT showed stenosis and oral-side dilatation of the small bowel (Fig. ), and he was diagnosed with small bowel obstruction. Although he was conservatively treated with fasting and infusion initially, the CT findings on hospital day 19 did not improve the bowel obstruction; therefore, an ileus tube was placed. The next day, gastrointestinal contrast examination using the ileus tube revealed at least two stenotic regions in the small bowel (Fig. ). Based on these findings, the patient was diagnosed with small bowel stenosis due to blunt abdominal trauma and received laparoscopic surgery on hospital day 21.\nFirst, a 12-mm port was placed using the open method in the umbilical site and the abdominal cavity was observed with a laparoscope. A localized area of the mesentery was congested and edematous, and scarring around the stenotic section of the small bowel was present (Fig. a, b). A small incision was made in the umbilicus, and the stenotic sections of the small bowel were brought outside through the abdominal wall. Close observation revealed three stenotic regions which were 40–50 cm from the Treitz ligament, and partial resection and anastomosis of the small bowel were performed. The operation time was 2 h and 20 min, and the blood loss was 5 mL.\nThe excised specimen showed three stenotic regions (Fig. ). The pathological examination revealed erosion of the mucosa and ulceration; the muscle layer was also destroyed and partially ruptured (Fig. a). The strong expansion and hyperplasia of the capillaries, which were recognized as findings of fibrosis, were also observed (Fig. b).\nThe postoperative course was stable, and the patient was discharged on postoperative day eight.
A 43-year-old man was admitted to a regional hospital (in Shahreza city) complaining of sudden onset of abdominal pain. Ultrasonography of abdomen and pelvis showed aneurysmal dilatation of distal part of abdominal aorta to be about 26 millimeters. The patient was sent to our hospital for further evaluation and treatment. He had pain attacks for 1-2 hours in periumbilical area radiating to all parts of abdomen. There was no relation between pain and position, eating, defecation and activity. The patient had no history of melena or limb neuromuscular dysfunction, but had a history of fever and chills for 2 days, which relieved spontaneously. The patient had no history of hypertension and diabetes mellitus but had a history of minor blunt abdominal trauma occurred 1.5 years ago. The vital signs were normal. In physical examination, no pulsatile mass was detected but a bruit was heard around the navel. Pulses in the lower extremities were normal. Chest X-ray, electrocardiography, echocardiography, complete blood count with diff and urinalysis were all normal but ESR was 65. All diagnostic tests of salmonellosis and other infectious or inflammatory diseases were negative. The patient had good urinary output. Spiral CT of abdomen and pelvis with IV contrast revealed infrarenal dissection aneurysm of abdominal aorta with extension to right common iliac artery () and aortography confirmed the diagnosis and revealed the treatment plan for the patient (). The size of the aorta was more than 5 cm in ultrasonography and CT scan performed in our center. In chest spiral CT, no dissection or aneurysm of thoracic aorta was detected (). Because of the severe abdominal pain (possibility of visceral ischemia) and large size of the aorta, midline laparotomy was done and a bifurcate collagen-coated Dacron graft was interposed between abdominal aorta and left common iliac artery and right common femoral artery (because the aneurysm was extended to the distal of right external artery). The patient was discharged after one week hospitalization and had no problems in 6 months follow-up.
The patient is a 52-year-old man with history of severe peripheral arterial disease status post amputation above the knee (AKA) on the left with a non-healing left stump wound and right lower extremity ulcerations. Patient has a long-standing history of type 2 diabetes mellitus and continues to smoke. He was referred for consideration of revascularization given his nonhealing wounds. Our institutional Review Board does not require informed consent or ethics approval for case reports.\nPatient underwent a peripheral angiogram from a right common femoral artery (CFA) access site. He was found to have a long occlusion from his mid to distal left external iliac artery into his CFA and proximal left superficial femoral artery (SFA). His right SFA also had a flush total occlusion. The right SFA occlusion extended to the distal portion of this vessel with reconstitution of the popliteal from the profunda femoris (PF). The right tibioperoneal artery was severely diseased and the posterior tibialis artery was totally occluded above the ankle. The right anterior tibialis was patent to the foot and had no significant disease. The proximal left external iliac artery underwent stenting from the contralateral approach at a site of severe 90% lesion at its origin. This vessel was feeding collaterals to his left stump. Patient returns in a few days for staged treatment of his right flush occluded SFA at its origin. Access site from the contralateral occluded left CFA was not possible. A dual access from the right radial artery and right dorsalis pedis (DP) artery was planned. Our case illustrates the feasibility of the radial access approach for peripheral angioplasty using long wires and balloons and shows the feasibility of the pedal-radial access for the treatment of a flush occlusion of a chronically occluded SFA.\nThe right DP was accessed using a micropuncture needle under ultrasound guidance. A 6F Glidesheath Slender (Terumo) was placed into the right DP. A total of 400 µg of intravascular nitroglycerin was administered via the pedal sheath. Also, a total of 4000 units of intravenous heparin was administered. The radial artery was then accessed with another 6F Glidesheath Slender and the long Wholey wire was advanced from the right radial sheath into the distal abdominal aorta over a 110 cm pigtail catheter. Abdominal aortogram was performed with iliac runoff noting flush occlusion of the right SFA at its origin and occlusion of the left external iliac and left CFA.\nUsing the Wholey wire, the short 6F Slender sheath was exchanged with a 118 cm long destination sheath (Terumo) placed in the distal aorta. The Wholey wire was then exchanged for a 400 cm 0.035″ stiff angled glidewire (Terumo) and introduced initially into the PF artery. A 150 cm glide catheter (Terumo) was then placed over the 400 cm wire and advanced to the right PF where an injection of contrast was performed to visualize the reconstitution of the right SFA and outflow vessels distally by collaterals (). The catheter was then pulled out into the right CFA for dye injection. The angled 150 cm NaviCross catheter (Terumo) was advanced from the pedal sheath with a 260 cm Glidewire Advantage (GWA) (Terumo) to cross the distal cap of the right SFA occlusion (). The pedal catheter and wire were advanced to the proximal SFA and the wire was noted to be in the subintimal space and could not cross into the right CFA despite the use of multiple wires (including 0.018″ Astato (Asahi) and V-18 (Boston Scientific) wires). The 400 cm wire from the radial approach was then able to go beyond the proximal cap with the aid of the NaviCross catheter and advanced intraluminally to the right popliteal. The monorail 200 cm length, 5.0 mm × 100 mm MetacrossRX balloon (Terumo) was then used to dilate the totally occluded right SFA. The V-18 wire was then advanced retrogradely from the pedal sheath into the true right CFA lumen into the distal aorta. Two 6 mm × 100 mm Zilver PTX (Cook) drug eluting stents were placed from the origin of the SFA to the medial SFA followed by a Lutonix (Bard) drug-coated balloons to the distal SFA. There was less than 5% residual narrowing seen in the SFA post treatment. The right CFA was then dilated with 6.0 mm × 60 mm Lutonix balloon for 3 min at 8 atm. A type C dissection was seen in the right CFA and an 8.0 mm × 80 mm Lifestent (Bard) was placed in the proximal 1/3rd and mid 1/3rd of right CFA via the pedal approach yielding 0% residual narrowing. The distal 1/3rd of the right CFA was not stented. Stents were post dilated with a 6.0 mm balloon (). The radial sheath was then pulled and hemostasis achieved with a TR wrist band (Terumo). The pedal sheath was removed and hemostasis was carried on with manual compression. Excellent pulses were seen in the right radial and right pedal post treatment.
Patient is a male, 77 years old, diabetic, and treated with oral hypoglycemic drugs and antihypertensive therapy, with good overall clinical condition. A mixed arterial and venous wide ulcer was localized in left ankle from three years. The patient has been followed up by an outpatient service of diabetes with topical treatments and advanced dressing with mixed results. The ulcer was extended over 50% of the circumference of the left ankle. The Achilles tendon was partially exposed. The patient was subjected to periodic Doppler ultrasound to check the venous and arterial circulation. The foot pulses were retained. The lesion was painful, in particular in the night hours. The ulcer was prepared by removing fibrin with a curette and the edges of the ulcer were freshened (). Local anesthesia has been practiced around the ulcer and in the donor site graft. Platelet rich plasma was prepared using gravitational platelet heparinazed system. Briefly, the patient's phlebotomization consisted of 27 mL of whole blood and was drawn from the median cubital vein with a 21-gauge needle. One 30 mL syringe with 3 mL of anticoagulant citrate dextrose (ACD) solution formula 3 cc of platelet-rich plasma was extracted. Holes were drilled in the ulcer using a punch 5 mm while maintaining a distance of one centimeter between the holes ().\nThe platelet-rich plasma has been infiltrated on the bottom and edges of the ulcer ().\nFrom the donor sites, skin was harvested with a 6 mm biopsy punch. The grafts were removed with scissors and tweezers and placed on sterile, saline-moistened cotton dressings; the punch grafts were taken from the arm. The grafts were placed in 5 mm holes arranged. Local anesthesia has been practiced in the ulcer compared to the donor site graft. The ulcer was medicated with hydrogel and a pressure dressing was removed after 8 days, in successive weeks the wound was medicated with paraffin gauze.\nIf the ulcers are very extensive, you can perform surgery in 2 or 3 steps ().\nAfter a few days the patient no longer reported pain. Granulation tissue appeared quickly between the grafts. Most of the grafts took root after 2-3 weeks and started the development of the islands of epithelial tissue that gradually came together to close the ulcer (). The process of reepithelialization was completed in four months (). The conditions of life of the patient are significantly improved by the absence of pain and the limited number of dressings.
A 58-year-old woman presented with symptoms of indigestion, belching and heartburn for 6 months. Initially, symptoms were intermittent and non-specific but later become associated with eating and affected her lifestyle, with upper abdominal pain with no relieving factors. She also started to develop infrequent vomiting, which tended to occur for a few times a week.\nShe has a medical history of hypertension and hypercholesterolemia and an open appendicectomy for acutely inflamed appendix. An abdominal ultrasonography scan showed gallbladder stones with a thick-walled gallbladder, but no signs of acute inflammation of the gallbladder.\nThe patient was subsequently seen in a general surgery clinic. She had an upper gastro-intestinal endoscopy to rule out any peptic ulcer disease, but it did not show any abnormality up to D2. The patient was offered a laparoscopic cholecystectomy to remove her gallbladder stones as a possible treatment for her symptoms and was booked as a day case.\nThe findings were a thick-walled gallbladder with adhesions to the omentum, transverse colon and stomach wall. On further dissection, we found that there was a dense adhesion between the gallbladder and the antrum of the stomach, and a provisional diagnosis of cholecystogastric fistula had been made.\nDue to technical difficulty to dissect laparoscopically, the operation had to be converted to open and a subcostal (Kocher) incision was created. The diagnosis of cholecystogastric fistula was confirmed with the finding of a fistula between the fundus of the gallbladder and the stomach antrum. The fistula was divided, the gallbladder resected by antegrade dissection method, the cystic artery and ducts identified and ligated and gallbladder was removed. The stomach fistula site was 5 mm in size, which was trimmed and then primarily repaired with two layers of vicryl 2-0 suture. The area was further covered by an omental patch to re-enforce the repair area. The patient postoperatively had uneventful recovery and discharged home after three days.\nThe patient was reviewed in outpatient after 10 weeks, and she has recovered properly with no postoperative complications.
A 37-year-old man was admitted to our hospital complaining of frequent episodes of headache for the past several months. He was followed by the otolaryngologist for sinusitis, but for the persistence of the headache, computed tomography (CT) and magnetic resonance (MR) of the brain were performed. CT scans showed a right frontal calcified lesion e () and marked enhancement post contrast and MR revealed hypointense lesion in the right frontal lobe with heterogeneous contrast enhancement after Gadolinium administration (). On admission to our department, general, physical and neurological examination found no abnormalities. X-ray scan of the skull revealed an extended geographical area of osteolysis in charge of the frontal squama in the right supraorbital region with involvement of the roof of the ipsilateral orbit and hypoplasia of frontal sinus. A brain CT and MR were repeated and showed no changes beyond those already performed. There were no stigmata or family history of neurofibromatosis.\nA right pterional approach was performed. The bone presented a gap full of blood sized 3 × 2.5 cm. The craniotomy was performed round this gap with a margin of 1 cm. The roof of the orbit ipsilateral was involved by the fibrous part of the lesion, which was not well demarcated from the surrounding soft brain parenchyma. We performed a progressive vascular deafferentation and disconnection of the lesion from the parenchyma to its anteromedial segment that appeared in proximity to the base of the superior sagittal sinus through the vein of Sperino. Dura appeared thickened, well vascularized and its dissection from the parenchyma was difficult. At the end, the alterated dural surface was replaced by a patch of galea and the bone reconstruction was obtained using his bony flap.\nHistological examination showed areas of collagen "dura-like" (clearly visible after staining with Masson's trichrome) accompanied by areas of bone metaplasia and presence of cerebral gray matter with normal architecture. There was a rich vascularization with vessels often with wall hyalinization, perivascular fibroblastic spindle cells and rare psammoma bodies. Coexisting areas, including extensive benign MA proliferation (EMA +), especially in the perivascular site, were present ().\nThe patient’s postoperative course was uneventful. At 6 months follow up, the patient reported no more episodes of headache (, ).
A 41-year-old male was admitted to the hospital with a major complaint of left knee pain that began 3-4 years ago. There was no specific history of trauma or infection on the left knee. The patient reported intermittent knee pain and feeling of irritation (catching) during flexion and extension of the knee that had been present for 3-4 years. Due to persistent pain and irritating feeling upon squatting and over 100° of flexion, activities involving knee flexion was impossible. Since 18 months ago, a mass was palpable on the skin in the medial side of the left knee, and it moved underneath the skin when pressed by the hand. When the mass was checked or palpated on the medial skin of the knee, the pain with locking worsened, whereas when it was not palpated after being pressed underneath the skin, the feature of the symptom was similar but the severity was relieved. On physical examination, a mass with the size of a bean was observed on the medial surface of the patella, and tenderness was present. Simple radiography did not demonstrate any shadow of mass or local bony erosion, and the joint space appeared normal.\nOn magnetic resonance imaging (MRI), the area where the medial plicae is present inferomedial to the patellofemoral joint showed equal intermediate-low signal intensity on T1 images and a 8×11×7 mm sized oval-shaped mass with a low signal intensity was found on T2 images (). On the contrast enhanced images, findings of specific enhancement and effusion of synovial fluid or synovitis were not present.\nOn arthroscopic examination, a yellowish oval shaped mass of pedunculated type protruding through the patellofemoral joint was found. No abnormal findings of hypertrophy of the medial plicae were found and the articular cartilage of the patella and femur was not injured. We made a high anterolateral portal as a viewing portal. En bloc resection of the mass was done through the anteromedial working portal (). The size of the resected mass was 14×10×11 mm and it was a yellowish solid tissue with clear margin, and the color inside was dark-brown ().\nIn hematoxylin and eosin stain, pathologic studies showed histiocyte, fibrous tissue, and giant cells. Multinucleated giant cells mixed in a collagen stroma and macrophage that has eaten hemosiderin were found. It was diagnosed as localized nodular tenosynovitis originating from synovium just above the medial plicae (). Pain and mechanical symptoms were relieved immediately after surgery. During the 1-year follow-up, no recurrence of preoperative symptoms or abnormalities was found in physical examination.
A 5-year-old girl was admitted to our Emergency Department after an episode of acute interscapular back pain occurring without trauma and followed by bilateral upper and lower limb weakness.\nHer family history included 8 Italian members harboring the same homoplasmic m.3460G > A mtDNA (Table , Fig. ). All the family members presented headache poorly responsive to NSAIDs and, except for the girl and her mother, visual loss due to optic nerve pathology. The patient’s medical history was unremarkable.\nThe patient’s vital signs were normal and stable. At the neurological examination, her cognitive functions were normal. There were no cranial nerve lesions. She was unable to stand and walk and had more severe weakness in the right lower limb. She also presented weakness in her upper extremities and difficulties with fine hand movements, particularly in the right hand. The right patellar and Achilles tendon reflexes were brisk. The plantar reflex was positive in the right foot. She presented impaired bladder control. No alterations in touch or pain sensitivity were present in the trunk and limbs. No concomitant signs of infection or inflammation were present, and no such signs had been reported in the previous weeks. Analyses of CSF pressure, glucose, protein, cell count, viral PCR, and culture were normal, and oligoclonal bands were absent (see timeline of events and treatment in Fig. ). The patient underwent a spinal cord MRI; the results showed hyperintense signal alterations in T2-weighted sequences and restricted diffusion in diffusion weighted imaging (DWI) sequences in the anterior portion of the cervical and dorsal spinal cord, suggesting anterior spinal artery territory involvement (Fig. ). Computed tomography angiography (CT) imaging showed no arterial dissection or other vessel abnormalities. Visual evoked potentials were normal. Somatosensory evoked potentials in both legs showed decreased conduction velocities. Motor evoked potentials showed lower amplitude for cortical derivation, prolonged latency in the upper limbs and normal in the lower limbs. Central conduction time was increased in the upper limbs and normal at the lower limbs. Complete autoimmunity and thrombophilia screening were unremarkable. Testing for anti-AQP4 antibodies was negative, and anti-MOG antibodies were not significantly elevated. A cardiologic consultation and echocardiography identified normal heart and aorta features. At the ophthalmological assessment, pupillary reactions were normal, the fundus oculi did not reveal pathological signs, and color vision was not affected. The visual acuity was 9/10 bilaterally. The digital visual field test displayed a mild defect in the peripheral portion of the visual field that was more evident in the left eye. Optical coherence tomography, visual evoked potential and electroretinograms were normal. As the child grows, her visual function will require careful monitoring, particularly when she reaches the adolescence.\nAfter obtaining written informed consent consistent with the principles set forth in the Declaration of Helsinki, total DNA was isolated from the patient’s peripheral blood according to standard protocols. Full-length mitochondrial DNA sequencing was conducted according to a previously described protocol [], and detected the haplogroup H27 was detected in the proband. The following variants were detected in the homoplasmic state: m.41C > T, m.73A > G, m.263A > G, m.1438A > G, m.3460G > A, m.4769A > G, m.8860A > G, m.11719G > A, m.15326A > G, m.16129G > A, m.16316A > G, m.16519 T > C. Based on the presence of the m.11719G > A and m.16316A > G nucleotide variants, the patient is predicted to belong to the H27 haplogroup. The m.3460G > A transition, a major mutation associated with LHON, was verified in blood-derived DNA of other family members by PCR-RFLP analysis.\nAcetylsalicylic acid was administered at low doses (2.5 mg/kg daily), and high doses of methylprednisolone (20 mg/kg daily) were empirically administered for three days. Although no proven treatments for LHON are available, early treatment with idebenone is thought to limit the progression of the disease; the patient was administered 45 mg of idebenone three times daily. Within two days following this therapy, clinical manifestations improved, and the patient regained bladder control and the ability to ambulate; within one week, the girl also recovered nearly normal strength in both arms. A control spinal cord MRI together with 1H-MRS to study the lactate peak was performed 10 days after hospital admission; the results showed complete regression of the alterations and no abnormal metabolites (Fig. ). The patient continued outpatient rehabilitation, and her motor functions improved, resulting in an almost completely normalized neurological examination after 2 months and preserving these achievements at follow-up one year later.
A 32-year-old female, gravida 0, presented to our hospital with a chief complaint of primary infertility. Her birth and peri-neonatal periods were uneventful, and she experienced menarche with regular menstruation lasting 7 days every 26-31 days. Her medical history included diagnosis of left ovarian endometriosis and she had taken low dose contraceptives. After she married, she had visited another hospital with a complaint of infertility, and she was referred to our hospital for treatment. Physical examination showed normal female physique and external genitalia. Speculum examination revealed a 7 cm blind vaginal canal of normal caliber with two bilateral 2-3 mm pinpoint perforating holes and no visualization of the cervix []. Transvaginal ultrasonography and MRI showed a normal cervix and uterus and 2.5 cm left ovarian endometrioma, but the septum of the vagina was not detected []. The urinary system was normal.\nThese findings led to suspected perforated TVS of the upper vagina or proximal vagina atresia, and surgical evaluation and treatment were performed under general anesthesia. With the patient placed in a dorsal lithotomy position, Foley catheters were inserted into the two apertures, 8 French to the left and 14 French to the right, and balloons were inflated with 3 ml and 8 ml of normal saline to fix each catheter in the proximal vagina []. About 30 ml of saline was injected through the catheters, and the proximal vaginal pouch distended with saline and the uterine cervix were detected by transabdominal ultrasound []. The Foley catheters were pulled down to prevent water leakage via the perforation of the septum during injection. The findings from saline infusion sonocolpography provided convincing evidence of perforated TVS of the upper vagina, rather than proximal vaginal atresia. To confirm the vaginal pouch and uterine cervix visually, the left Foley catheter was withdrawn and a flexible endoscope was inserted into the vaginal pouch, which revealed a completely normal portio []. Saline infusion sonocolpography revealed that the septum was 3 mm thick and created the proximal vaginal pouch. The septum was excised with cautery and a completely normal cervix was ascertained on speculum examination []. Postoperatively, self-dilation of the vagina was introduced with a dilator made of polyethylene (length 10 cm; diameter 3 cm) for 20 minutes every day for 2 months to prevent agglutination of the excised area. The postoperative course was uneventful.
A 72 year old man was admitted to our department with a nine month history of gradually enlarging mass in the volar aspect of the hypothenar area of his left hand. The patient reported that the size of the mass had been increasing during the past 3 months. As far as the rest medical record is concerned he was under medication for diabetes mellitus type 2 with metformin and he had undergone an operation for colorectal malignancy 6 years before presentation. He had no previous hand injury. The patient experienced persistent pain and altered sensation on the ulnar side of the hand, accompanied with progressively diminishing grip strength. Physical examination revealed a small, mobile, and painless palpable mass in the hypothenar area. The skin overlying the mass was normal. The range of motion of the finger joints was not restricted and there was no vascular impairment.\nLaboratory exams were within normal limits and radiographs of the hand demonstrated no osseous involvement or radio-opaque lesion. Ultrasound (U/S) examination revealed a solid, hypoechoic and homogeneous tumor with capsule in the ulnovolar aspect of the left hand (). Magnetic Resonance Imaging (MRI) indicated a multi-lobulated mass, located in the volar aspect of the fourth web space of the hand, not infiltrating adjacent structures or having bony involvement and bone edema (). No biopsy was performed preoperatively, since all radiological studies suggested a benign tumor.\nThe patient was operated under axillary nerve block. Using a Brunner incision volarly the skin and the palmar fascia were divided carefully. The neurovascular bundles were identified and protected. The mass was removed en block with clear margins. The gross specimen consisted of a lobulated, well-circumscribed, gelatinous mass ().\nThe histopathology of the specimen revealed () uniform, cytologically bland spindle-shaped cells with eosinophilic cytoplasm separated by abundant extracellular myxoid stroma containing very sparse capillary – sized blood vessels.\nAfter the last follow up, 12 months following the excision, the ring and the little finger of the patient had normal sensation and range of motion (). No signs of tumor recurrence were noticed during follow up.
An 11-year-old boy presented with an extensive comminuted open fracture of the left elbow associated with massive primary bone loss, measuring more than 10 cm in length. The patient lost the distal articular surface of the humerus, a 7 cm segment of the ulnar nerve, and ended up with a soft tissue defect over the medial elbow measuring about 7 by 10 cm. Other nerves and major vessels were intact. Limited internal fixation and an external fixator were used prior to his referral to our institution. The fixator pins were placed in the radius instead of the ulna, and the forearm was in neutral pronation. He arrived 3 weeks after the injury ( and ). The radiographs showed subtotal articular cartilage loss, an injured distal humerus growth plate, and missing olecranon apophysis. Computerized tomography angiography revealed patent brachial, radial, and ulnar arteries. To ensure a sterile surgical bed, the child underwent several trips to the operating room for debridement and placement of antibiotic beads. Intraoperative tissue cultures were done because of late presentation and grew Enterobacter cloacae. Broad-spectrum antibiotics were initially started and then changed to culture-specific antibiotics that were administered for 40 days. Once the pouch was clean, PMMA block mixed with gentamicin was placed as the first stage of the induced membrane procedure and covered with a pedicled radial forearm flap and skin graft. The original fixator was left in place.\nThree months later, the soft tissue had healed, and serum inflammatory markers were normal. Therefore, treatment proceeded to the second stage. The flap was lifted followed by identification of the ulnar nerve stumps. The membrane was sharply opened, and the thick membrane was preserved. We then removed PMMA piecemeal. To fill the void, 50 cm of ipsilateral iliac crest morselized cancellous autograft mixed with 45 cm of allograft chips and 10 cm of demineralized bone matrix was added. The cavity was closed watertight in a running fashion (). For long-term stability after removal of the fixator, we placed a spanning locking reconstruction plate medially. Based on the child and his family preference, we kept the flexion at 65°. The ulnar nerve gap was reconstructed with sural nerve grafts. The patient was observed for another 11 months before we removed the fixator after consolidation of the transplanted bone. The patient’s wounds healed without surgical-site incidents.\nDuring the follow-up assessment 30 months after the injury, the patient was satisfied with the appearance of the limb and had no pain or limitations when performing activities of daily living. There was mild residual clawing of the ring and little fingers, but near symmetric grip strength. He had partial recovery of the ulnar nerve. Radiographs confirmed healing ().
A 54-year-old female patient visited our pain clinic and reported a 6 month history of severe left inguinal, buttock and posterior thigh pain; the pain started from the inguinal area and spread to the other regions gradually. The pain had begun with the accident which abruptly raised the frequency and amplitude of TENS, which had been being applied to her left knee, by another patient in the next bed. She described the pain as intermittent and intense, with a feeling like it was being pulled with a jerk, and that it was aggravated by hip joint motions. Her numerical rating scale (NRS) score for pain (0 means no pain, 10 means the worst pain imaginable) was 8-9. And she had previously had a paroxysmal tremor of the left leg when the pain was the most intense. She had undergone right anterior cruciate ligament reconstruction 1 month prior to the TENS accident. And the TENS was applied to treat the allodynia and hyperalgesia after knee surgery. Her right knee pain disappeared by itself during the treatment for the left-side pelvic pain.\nThere was no abnormal focal neurologic sign. And she underwent a series of investigations, including blood tests and imaging studies, which included the lumbar spine, pelvis and hip joint MRI, none of which identified the cause of her pain. We tried to perform electromyography (EMG) and nerve conduction studies (NCS), but failed to complete these examinations due to severe pain attacks provoked by insertion of the EMG needle. Severe tenderness was noticed in the gluteus minimus on palpation. Because the range of motion (ROM) test and palpation provoked severe pain, it was hard to examine other muscles of the pelvic girdle at her first visit. On the initial impression of myofascial pain from the gluteus minimus, we injected 10 ml of 0.1% ropivacaine into the muscle under ultrasound guidance.\nAt her second visit, we identified that there was a 30% improvement of buttock pain and about a 10% improvement in inguinal pain. On physical examination, there was severe pain and tenderness around the pubic tubercle and lesser trochanter, especially when she actively flexed her hip joint with knee extension in the supine position. On manual muscle testing, motor weakness defined by an MRC grade of 4 was noted during the left hip flexion and adduction. To rule out a neuropathy of a peripheral nerve such as the lumbar plexus or obturator nerve, we tried again to perform EMG/NCS while administrating fentanyl via the intravenous route. But there was no definitive electrodiagnostic abnormality of the lumbar plexus or obturator nerve. We assumed that her pain might result from the iliopsoas or adductor muscles although we could not identify a trigger point or taut band probably due to a hypertonic muscle with severe pain. We performed a fascia iliaca compartment block under ultrasound guidance and the inguinal pain was relieved immediately. However, her pain relapsed 1 week later without further improvement on repeated blocks. Therefore, we injected 10 ml of 0.1% ropivacaine on the distal belly of the iliopsoas around the lesser trochanter under fluoroscopic guidance. On the next follow up visit, we identified an improvement of inguinal pain of more than 50%, thus allowing palpation of the muscles supporting the hip joint and pelvis. Severe tenderness and a jump sign was provoked when palpating the pubic tubercle and superior and inferior pubic rami. Therefore, we performed pectineus and obturator externus muscle injections around the pubic tubercle and rami using 5 ml of 0.1% ropivacaine under fluoroscopic guidance, respectively.\nAt the next visit, the patient did not complain about the inguinal pain, but presented severe buttock and posterior thigh pain with an intermittent tremor on her left leg. On physical examination, severe tenderness of the piriformis was noted and a paroxysmal left leg tremor developed during passive internal rotation of the hip in prone position. We performed a piriformis muscle injection using 10 ml of 0.1% ropivacaine under ultrasound guidance. After the injection we saw an immediate resolution of the tremor. When the patient returned to our pain clinic one week after the last treatment, the pain in the groin, buttock, and posterior thigh was much relieved as reflected in the NRS score of 1-2. At 1 the month follow up visit, the patient continued to be free of pain with mild discomfort on hip flexion.
A 5-year-old girl was admitted to our Emergency Department after an episode of acute interscapular back pain occurring without trauma and followed by bilateral upper and lower limb weakness.\nHer family history included 8 Italian members harboring the same homoplasmic m.3460G > A mtDNA (Table , Fig. ). All the family members presented headache poorly responsive to NSAIDs and, except for the girl and her mother, visual loss due to optic nerve pathology. The patient’s medical history was unremarkable.\nThe patient’s vital signs were normal and stable. At the neurological examination, her cognitive functions were normal. There were no cranial nerve lesions. She was unable to stand and walk and had more severe weakness in the right lower limb. She also presented weakness in her upper extremities and difficulties with fine hand movements, particularly in the right hand. The right patellar and Achilles tendon reflexes were brisk. The plantar reflex was positive in the right foot. She presented impaired bladder control. No alterations in touch or pain sensitivity were present in the trunk and limbs. No concomitant signs of infection or inflammation were present, and no such signs had been reported in the previous weeks. Analyses of CSF pressure, glucose, protein, cell count, viral PCR, and culture were normal, and oligoclonal bands were absent (see timeline of events and treatment in Fig. ). The patient underwent a spinal cord MRI; the results showed hyperintense signal alterations in T2-weighted sequences and restricted diffusion in diffusion weighted imaging (DWI) sequences in the anterior portion of the cervical and dorsal spinal cord, suggesting anterior spinal artery territory involvement (Fig. ). Computed tomography angiography (CT) imaging showed no arterial dissection or other vessel abnormalities. Visual evoked potentials were normal. Somatosensory evoked potentials in both legs showed decreased conduction velocities. Motor evoked potentials showed lower amplitude for cortical derivation, prolonged latency in the upper limbs and normal in the lower limbs. Central conduction time was increased in the upper limbs and normal at the lower limbs. Complete autoimmunity and thrombophilia screening were unremarkable. Testing for anti-AQP4 antibodies was negative, and anti-MOG antibodies were not significantly elevated. A cardiologic consultation and echocardiography identified normal heart and aorta features. At the ophthalmological assessment, pupillary reactions were normal, the fundus oculi did not reveal pathological signs, and color vision was not affected. The visual acuity was 9/10 bilaterally. The digital visual field test displayed a mild defect in the peripheral portion of the visual field that was more evident in the left eye. Optical coherence tomography, visual evoked potential and electroretinograms were normal. As the child grows, her visual function will require careful monitoring, particularly when she reaches the adolescence.\nAfter obtaining written informed consent consistent with the principles set forth in the Declaration of Helsinki, total DNA was isolated from the patient’s peripheral blood according to standard protocols. Full-length mitochondrial DNA sequencing was conducted according to a previously described protocol [], and detected the haplogroup H27 was detected in the proband. The following variants were detected in the homoplasmic state: m.41C > T, m.73A > G, m.263A > G, m.1438A > G, m.3460G > A, m.4769A > G, m.8860A > G, m.11719G > A, m.15326A > G, m.16129G > A, m.16316A > G, m.16519 T > C. Based on the presence of the m.11719G > A and m.16316A > G nucleotide variants, the patient is predicted to belong to the H27 haplogroup. The m.3460G > A transition, a major mutation associated with LHON, was verified in blood-derived DNA of other family members by PCR-RFLP analysis.\nAcetylsalicylic acid was administered at low doses (2.5 mg/kg daily), and high doses of methylprednisolone (20 mg/kg daily) were empirically administered for three days. Although no proven treatments for LHON are available, early treatment with idebenone is thought to limit the progression of the disease; the patient was administered 45 mg of idebenone three times daily. Within two days following this therapy, clinical manifestations improved, and the patient regained bladder control and the ability to ambulate; within one week, the girl also recovered nearly normal strength in both arms. A control spinal cord MRI together with 1H-MRS to study the lactate peak was performed 10 days after hospital admission; the results showed complete regression of the alterations and no abnormal metabolites (Fig. ). The patient continued outpatient rehabilitation, and her motor functions improved, resulting in an almost completely normalized neurological examination after 2 months and preserving these achievements at follow-up one year later.
Here, we are presenting a rare case of a 11-year-old boy referred to our Department of Pediatric and Preventive Dentistry with the chief complaint of pain and swelling on the lower left back tooth region from the past 20 days (). Swelling was initially small in size gradually progressed to present size and associated with extraoral draining sinus for which he consulted a private dental clinic. Although he was under medication for past 1 week, there was no reduction in the size of the swelling (). His medical history revealed multiple fractures sustained during routine handling with difficulty in walking. His birth and neonatal period were uneventful. Family history revealed a positive finding of the same illness in his elder sibling. On examination, child was sick looking, pale, dyspneic, with rhizomelic dwarfism, and relative macrocephaly with frontal bossing. There was bilateral bowing of lower limbs with varus deformity and flexion deformity of the thumb. Patient profile was concave with typical triangular faces with fragile skin (). He had white sclera with narrow thoracic cage. Abdomen was protuberant with marked kyphosis. Patient had undergone surgery for impaired vision and hearing was normal. There was marked scoliosis with convexity toward left. Asymmetry was seen on the left side of the face with palpable submandibular lymph nodes, which was mobile and tender on palpation. On inspection, a solitary diffuse swelling was present on the left side of the face measuring 2 × 3 cm, extending from the corner of the mouth to middle third of the mandible anteroposteriorly, and from ala-tragal line to the base of the mandible superior-inferiorly, associated with draining sinus. The swelling was soft in consistency, fluctuant, mild-tender on palpation, and there was a localized rise in temperature. On intraoral examination, swelling present in relation to 75 extending from attached gingiva up to buccal vestibule (). Deep dentinal caries was present in relation to mandibular left primary second molar which was tender on percussion and presented with vestibular tenderness and obliteration. The intraoral lesion was rubbery and fluctuant on palpation. Tooth present was 15 53 12 11 21 22 25 84 42 41 31 32 75. Dental caries with respect to 11, 21, 15 and 25, caries with pulp exposure with respect to 75, 84. Intraoral periapical radiograph and orthopantomograph and cone-beam computed tomography were taken (), and finally, diagnosis has arrived as dentoalveolar abscess with respect to 75 associated with extraoral draining sinus and pathological fracture of body of mandible on the left side. Investigation with hemogram revealed microcytic hypochromic anemia with relative lymphocytosis, echocardiography showed mild pulmonic regurgitation renal function test, and electrolytes were normal. Reduced Serum calcium level. Alkaline phosphatase was significantly raised, bilateral pneumonitic patches and haziness with lack of air spaces seen in chest X-ray. Hip X-ray revealed protrusion acetabulum. X-ray of the extremities showed thinned cortices ( and ). X-ray spine shows platyspondyly. Owing to his physician's high index of suspicion, a diagnosis of OI-III was pursued, which is the non-lethal form of the disease.\nCalcium and vitamin D therapy was initiated, and an in-depth discussion regarding bisphosphonates was pursued. Advised oral prophylaxis and oral hygiene instructions, lesion sterilization and tissue repair were done in relation to 75, pulp therapy with respect to 84, and restoration in relation to 15, 11, 21, and 25 were done. Tablet Augmentin 375 mg, Soframycin ointment, and tablet Metrogyl–200 mg were prescribed for 5 days. Patient was recalled after 3 days, 10 days, 15 days, 1 month, 3 months, and 4 months. Clinically healing extraoral sinus was noticed, and intraorally the findings were satisfactory and erupting 43 was noted ().
A 35-year-old woman presented to the outpatient department with complaints of bilateral nasal obstruction for 1 month associated with intermittent blood stained nasal discharge. The patient also gave history of swelling over the dorsum of the nose which developed rapidly over 1 month. There was no history of headache, facial pain, or visual disturbance. There was history of similar swelling in the past over the same site for which she had undergone a minor surgery 5 years back at a different hospital and the records of the biopsy were untraceable.\nOn examination, there was a firm, fixed, and well-defined swelling of size 2 × 2 cm over the root of the nose in the midline. On nasal endoscopy, left nasal cavity showed a fleshy mass filling the entire cavity pushing the septum to right lateral nasal wall.\nContrast-enhanced computed tomography of the nose and paranasal sinuses showed a heterogeneously enhancing soft-tissue density over root of the nose with erosion of nasal bone and extension into left nasal cavity and filling left maxillary sinus, anterior, and posterior ethmoid sinuses and extending into right nasal cavity with erosion of bony septum (. There was no skull base or lamina papyracea erosion or any enlarged neck nodes. Fine-needle aspiration of the external nasal swelling and biopsy from the nasal mass showed scattered pleomorphic epithelial cells having hyperchromatic nuclei and scanty cytoplasm with 1 to 2 mitotic figures per high-power field, surrounded by abundant basophilic chondromyxoid stroma suggestive of malignant chondroid syringoma. Patient underwent whole-body positron emission tomography–computed tomography to rule out metastasis in lungs, liver, and skeletal system which failed to show any uptake in the site other than nose and paranasal sinus region.\nPatient underwent excision of the mass by external approach (lateral rhinotomy incision) along with left medial maxillectomy and excision of the external subcutaneous component (). Postoperative biopsy confirmed the diagnosis of malignant chondroid syringoma. Patient received intensity-modulated radiotherapy 60 Gy/30 fractions, 4 weeks after the surgery. Patient was followed up every month in the first 6 months with diagnostic rigid nasal endoscopy, followed by 6 monthly interval for 2 years and found to have no further recurrence.
A 26-year-old female was admitted to the Department of General Surgery of Daping Hospital and Research Institute of Surgery (Chongqing, China), due to the presence of a painless mass in the left breast for three months. The patient indicated that the mass had recently grown rapidly. The patient had no notable medical history or family history of carcinoma. Clinical examination revealed a 3×2-cm firm irregular mass in the upper inner quadrant of the left breast. There was no change in the appearance of the local skin, no discharge from, or retraction of, the nipple. A small number of lymph nodes were palpated in the left axilla. The breast magnetic resonance imaging result indicated left breast cancer due to the presence of enlarged left axillary lymph nodes. A chest computed tomography (CT) scan demonstrated widespread lung and pleural nodules, indicating lung and pleural metastases. An emission CT whole body bone scintigraphy indicated destruction to multiple ribs, the cervical vertebrae and thoracic bone. A core needle biopsy of the breast mass and hematoxylin and eosin staining demonstrated that the mass tissue was comprised of a large distribution of diffuse small cells. Those cells were round or oval, with large nuclei and nucleoli, and abundant cytoplasm. No significant intracellular pigmentation was observed (). Immunohistochemistry demonstrated that the tumour cells were immunopositive for S-100, HMB-45 and melan-A (). However. a panel of markers that included epithelial markers, such as cytokeratin (CK) and epithelial membrane antigen (EMA), and mesenchymal markers, such as vimentin, smooth muscle antigen (SMA), estrogen receptor, progesterone receptor and HER2 were negative. The percentage of Ki-67-positive cells was 30%.\nBased on the pathologic and immunohistochemical features, a diagnosis of malignant melanoma was proposed. Careful examination of the skin and mucous membranes failed to reveal a malignant melanoma. Therefore this patient was diagnosed with a primary malignant melanoma of the left breast with extensive metastasis. The patient refused surgery and further treatment and was automatically discharged. Two months later, the patient succumbed as a result of widespread metastases.
A 54-year-old Japanese woman visited our ophthalmology department after experiencing proptosis, lid swelling, diplopia, and retro-orbital pain in her left eye lasting for 1 day. She had a medical history of poorly differentiated adenocarcinoma of the stomach, which had metastasized to her ovary and mesentery, diagnosed 2 years earlier. She had undergone four regimen courses of chemotherapy, yet these had failed and she thus received palliative treatment. There were metastases to subcutaneous tissue of her neck and thoracic bone marrow 3 months before her initial visit to our ophthalmic department. She had been admitted to our hospital 5 days previously without symptoms in either eye. She had undergone stenting in her esophagus against eating difficulties but she lived a self-reliant life at home.\nAt her first visit, an external examination showed lid swelling, red coloration, and proptosis of her left eye. A motility examination revealed an adduction deficit of − 4.0 and an abduction deficit of − 1.0. Ophthalmological examinations revealed a best-corrected visual acuity of 20/20 and an intraocular pressure of 15 mmHg in both eyes. No abnormal findings were found in the anterior segment. Her pupils were equally reactive without any relative afferent pupillary defect. A funduscopic examination showed partial optic disc edema in her left eye (Fig. a). No choroidal masses or striae were noted.\nA CT scan performed 10 days before her initial visit to our ophthalmology department revealed enlargement of the left medial rectus muscle. Retrospectively, similar findings were seen on a CT scan performed 3 months previously, and had worsened in the interim. Yet, a CT scan that had been performed 6 months previously showed no remarkable findings (Fig. ). There was no enlargement of other extraocular muscles and no swelling or mass lesion in other orbital tissues during the 6 months. So, gastric cancer metastasis to the medial rectus muscle of her left eye was suspected. Radiation therapy for metastasis to the subcutaneous tissue of her neck and thoracic bone marrow was effective; she received a total of 20 Gy/5 courses of radiation therapy to the orbit. A few days after completion of radiation therapy, lid swelling, red coloring, and pain disappeared. Two weeks post-radiation therapy, a motility examination revealed an adduction deficit of − 4.0 and Hertel’s exophthalmometry measurements with a 108-mm base were 14 mm (right eye) and 19 mm (left eye). At 1.5 months post-radiation therapy, a motility examination revealed an adduction deficit of − 2.0 and Hertel’s exophthalmometry measurements (108-mm base) were 14 mm (right eye) and 13 mm (left eye). A posterior ocular segment examination showed a normal left optic disc (Fig. b). She died 3 months after her initial presentation to our ophthalmology department.
A 38-year-old male presented to the surgery outpatient department with complaints of odynophagia and chest pain for two weeks. The symptoms had started after a meal when the patient felt the sensation of a foreign body in the throat, which he reportedly swallowed with a bolus of food. As the initial evaluation with neck and chest X-rays was reported as normal, he did not receive any specific intervention. However, in the third week after ingestion, he developed chest pain and fever. On evaluation with a contrast-enhanced thorax, chest, and abdomen scan, he was diagnosed with a right-sided pleural effusion and a suspected foreign body in the periesophageal region of the lower thoracic esophagus close to the inferior vena cava (Figures , ).\nFrom the hiatus region, the foreign body was located at approximately 5 cm cephalad. An intercostal drain was placed under image guidance and approximately 750 mL of serosanguinous pleural effusion was drained. An upper gastrointestinal contrast study did not show any contrast extravasation from the esophageal lumen. Upper gastrointestinal endoscopy also did not show any signs of recent perforation or a foreign body in the esophagus. An after adequate chest optimization, he underwent laparoscopic foreign body removal.\nThe procedure was done under general anesthesia in the supine split leg position. After initial access and the creation of a pneumoperitoneum through the left paramedian 12 mm port using an open method, five additional 5 mm ports were placed, including two midclavicular line ports on either side, a left subcostal port for assistance, and an epigastric port for liver retraction. The gastrohepatic ligament was divided to reach the right crus. The phrenoesophageal membrane was then opened to enter the mediastinum. The esophagus was then looped with an umbilical tape at the level of the hiatus and used for subsequent retraction. The right side of the esophagus was dissected meticulously, preserving the vagus nerves. At about 6 cm from the gastro esophageal junction, there was a fibrotic tract along the lateral aspect, which was then cut with scissors exposing the 2.7 cm long metallic foreign body (Figure ).\nRight pleura were thickened and were not opened. The foreign body was then extracted and removed under vision (Figure ).\nThe tract, which was seen extending to the esophagus, was clipped on the esophageal side using a hemolock. A thorough mediastinal lavage was given and a 16 Fr suction drain was placed in the paraesophageal region. An upper gastrointestinal oral contrast study that was done on the second postoperative day did not reveal any contrast leak, and the patient was started on an oral diet. He had an uneventful postoperative course and was discharged on the fifth postoperative day.
A 50-year-old Chinese man with a 5-year history of neck and shoulder pain presented with muscle atrophy and weakness in both arms. He was admitted to hospital with complaints of unstable holding for 9 months and numbness of limbs for 2 months, which worsened in one week. Five years previously, he tried the physiotherapy due to the pain of neck and shoulder, and relief from pain followed. Nine months previously, muscle atrophy and weakness in both arms were noted but without being promptly treated. And his pain of neck was obviously worsened 2 months before admission, together with numbness of limbs; the cervical and lumbar image examinations in other hospital were suggestive of protrusion of intervertebral disc at the C5–7 level and the L4-S1 level. Then he received the physiotherapy and felt the relief of pain, but numbness of limbs did not significantly relieve, which suddenly worsened in one week.\nThe patient also presented with a 3-year history of diabetes mellitus (DM), a 20-year history of gout involving left wrist and left knee, a 30-year history of smoking and drinking. Previous pharmaceutical treatment for gout had been intermittent for about 1 year, consisting of dexamethasone for acute episodes. Patient’s family and psychosocial history were not relevant for this case report.\nAfter admission, physical examination was performed in accordance with best practice and clinical guidelines [] by physician, in which multiple tophi were seen in left wrist, both feet and knee; bilateral superficial sensory declined below the level of mastoid portion, particularly below the knee, which presumably resulted from the lumbar lesions. The muscle strengths of upper limbs were graded 2+ to 4 and lower limbs were graded 0 in accordance with Medical Research Council (MRC) Scale for Muscle Strength []. The reflex of bilateral biceps, triceps and radial periosteum disappeared; the reflex of bilateral knee was normal and that of bilateral ankle disappeared. The muscle atrophy of bilateral thenar, hypothenar, forearms, biceps, triceps deltoid and quadriceps femoris, triceps surae was also noted. The gout history and physical examination revealing tophi in multiple joints informed the physician to perform some biochemical tests relating to gout, which found hyperuricemia with a level of 542 umoles/L (normal range, 208–428 umoles/L), and the C-reactive protein was 125 mg/L (normal range, 0–8 mg/L).\nBased on the symptoms and physical examination, physician intended to consider peripheral neuropathy and could not rule out cervical spondylotic myelopathy. Then the cervical computed tomography (CT) and contrast enhanced magnetic resonance imaging (MRI) were performed. CT scan showed the slightly irregular osteolytic destruction of the C5–6 endplates with discontinuous peripheral sclerotic margins and multiple small calcification and sequestra. Narrowing of C5–6 disc space and mild spinal stenosis were also found (Fig. ). MR images displayed diffused abnormal signal of the C5 and C6 vertebrae marrow, which were low signal intensity on T1-weighted images and intermediate to high signal intensity on T2 and short time inversion recovery (STIR) images. The erosion and destruction of both adjacent C5–6 endplates showed discontinuous and waved changes. The C5–6 intervertebral disc appeared fluid like signal changes that were hypointense on T1 and hyperintense on T2-weighted images. Swelling of prevertebral soft tissue was also found. Contrast-enhanced images showed significant enhancement of the C5–6 vertebrae marrow with no enhancing intervertebral disc. Spinal dural thickening and enhancement of the C5–6 level could be also seen on the enhanced T1-weighted images (Fig. ).\nThe timeline for the development of this case is summarized in Fig. .\nIn this case, with clinical features, laboratory tests and imaging studies, physicians initially had difficulty in determining the lesions of C5–6; both infectious spondylodiscitis and spinal gout could not be ruled out. In terms of laboratory tests, it was difficult to judge that hyperuricemia was caused by peripheral gout or new lesions, and that the elevation of C-reactive protein was caused by gout or infection. As for the imaging studies of low specificity, radiologist was not completely sure of the etiology and such case was rarely met at that time; initially the imaging reported the first consideration was infectious spondylodiscitis. Under the circumstance of the lesions being not determined and nerve root symptoms, surgical treatment was performed, for the purpose of timely decompressing the spinal cord and providing conditions for the recovery of its function. Intraoperative findings showed that intervertebral disc of C5–6 was completely destructed and replaced by turbid fluid and the erosion of adjacent endplates and vertebrae. The pathological examination of the specimen demonstrated deposited uric acid crystals surrounded by granulomatous inflammation, as well as fiber and angiogenesis with a large number of chronic inflammatory cells infiltration (Fig. ). Postoperatively, the lesions of C5–6 were definitely diagnosed as gout.\nPreoperatively, pharmaceutical treatment was given to relieve pain, nourish nerve and dehydrate []. To immediately decompress the nerve root symptoms, surgical treatment including anterior cervical corpectomy, debridement and fusion, and internal fixation of C5–6 was performed [, , ]. After surgery, the muscle strengths of limbs were significantly improved. Postoperatively, the new diagnosis was confirmed; then colchicine and prednisolone were respectively given 1 mg t.i.d and 10 mg t.i.d. whose dosage reduced when symptoms improved [, ]. Rehabilitation training including electronic biofeedback therapy, pneumatic compression, equipment training and electronic standing bed training was also performed [].\nAbout 3 months after surgery, the patient felt that pain of neck and shoulder was not obvious and there was no sensory abnormality in the upper limbs; the muscle strengths of limbs were all improved, with upper limbs grade 4 and lower limbs grade 2- to 4 in accordance with Medical Research Council (MRC) Scale for Muscle Strength []. The level of serum uric acid was well controlled between 336 umoles/l and 399 umoles/l (normal range, 208–428 umoles/l).
A 79-year-old male with longstanding history of advanced prostate cancer on androgen deprivation therapy presented to his urologist after noticing a mass on the tip of his urethral meatus. A subsequent biopsy of the mass was positive for SCC, and the patient underwent partial penectomy and lymph node dissection that revealed positive right inguinal lymph nodes (three out of seven) revealing pathologic T2N2M0 disease. He received adjuvant chemotherapy by extrapolating data of its benefit when given in the neoadjuvant setting. The standard TIP regimen was not pursued given patient's concern for side effects. The patient proceeded with alternative plan of chemoradiation with 5 weeks of weekly low dose carboplatin and paclitaxel. In addition, he received radiation with a total dose of 5000 cGy over 25 fractions to the right inguinal region. However, the patient developed disease recurrence with nodal involvement nine months later. On restaging CT imaging, the patient was found to have new involvement of the left pelvis. A nodal conglomerate measuring 31×58 mm with central necrotic change was identified in the left inguinal region.\nGiven the patient's age, performance status, and local recurrence of disease, he was started on therapy with chemoradiation with curative intent one month later. Treatment with an additional round of chemoradiation with low dose carboplatin and paclitaxel was given for 5 weeks. He had radiation with a total dose of 5000 cGy over 25 fractions to the left pelvic region. He had stable disease with chemoradiation, but he eventually developed disease progression within a year from the end of chemotherapy. At that point, he was considered for second-line therapy with the PD-L1 inhibitor atezolizumab. After being on atezolizumab for approximately 2 years, he developed biopsy-proven bullous pemphigoid, an immune-mediated toxicity of the skin that has been described with those agents. A restaging scan at approximately 2 years showed near complete response, so patient has been placed on treatment holiday at the time of this report. He was started on prednisone 1 mg/kg per immune-mediated management guidelines and had quick resolution of his blistering symptoms [].
A 66-year-old woman underwent screening mammogram in October. During the screening mammogram, the patient mentioned a lump in the lower inner quadrant of her left breast. The mammogram did not show any abnormal finding in this palpable area; however, the mammogram did show an approximately 6 mm mass in the upper outer quadrant of the left breast []. This lesion was further evaluated with a targeted ultrasound. Ultrasound demonstrated an irregular hypoechoic mass at 2 O’clock position [], corresponding to the abnormality seen on the mammogram. Given the suspicious appearance of the mass, ultrasound-guided core biopsy was performed.\nHistologic sections showed a cellular neoplasm arranged in vague lobules with delicate fibrovascular cores embedded in a fibrous stroma []. The tumor cells were uniform and bland with a moderate amount of granular eosinophilic cytoplasm. The nuclei were round to oval and uniform with granular chromatin and absent nucleoli. The morphologic differential diagnosis included solid papillary carcinoma of the breast versus metastatic WDNET.\nImmunohistochemical stains showed the tumor cells were positive for synaptophysin, supporting neuroendocrine differentiation, which could be seen in either lesion. Importantly, the tumor cells were negative for estrogen receptor arguing against solid papillary carcinoma of the breast. In addition, the tumor cells were positive for caudal-related homeobox gene 2 and somatostatin receptor Type 2A. The immunohistochemical profile, in conjunction with the tumor morphology and the imaging findings, was most compatible with metastatic midgut WDNET.\nBreast MRI was also performed to evaluate the extent of the disease which demonstrated the irregular enhancing mass in the upper outer quadrant of the left breast [], corresponding to the known biopsy proven metastatic neoplasm. MRI identified additional lesions in the liver as well as Level 2 lymphadenopathy seen between the pectoralis major and minor muscles (Rotter's node) [].\nSubsequently, the patient underwent to a CT of the abdomen and pelvis which showed at least seven liver lesions, with the largest lesion at segment 4 measuring approximately 2.6 cm []. There was also a spiculated mass with calcifications in the right lower quadrant near the ileocecal valve concerning for primary ileal WDNET []. The patient then underwent OctreoScan [] which demonstrated increased uptake in the left axillary Level 2 lymph node, within the breast lesion itself, in the liver lesions, and within the ileal mass consistent with primary ileal WDNET with metastasis to the breast, liver, and axillary lymph node. Fine needle aspiration of the left Level 2 axillary lymph node was performed, and the cytology was consistent with metastatic WDNET.\nOn further questioning, the patient reported night sweats, heat intolerance, episodes of lightheadedness, some stomach pain, and constipation, consistent with symptoms of ileal WDNET with hepatic metastasis.\nA treatment plan including ileal resection, cholecystectomy, and liver ablation with resection of the pectoralis lymph node as a separate operation was offered to the patient. The patient was very reluctant to pursue any operative treatment at this time.
The patient is a lean 6-year-old boy with a history of celiac disease who was diagnosed with type 1 diabetes mellitus and started on a continuous glucose monitor. Five months later, his parents placed a new sensor in the right lower quadrant abdominal wall. They did not receive a signal from the sensor and removed it, but noticed the wire had detached. By the following week, pain, swelling, and redness were noted over the site. An X-ray revealed the retained wire within the abdominal wall at the insertion site. An elective removal of the wire under sedation was scheduled with pediatric surgery. Prior to removal, the parents reported placing another sensor from the same box into the abdominal wall in the left lower quadrant. Once again, they did not receive a signal from the sensor, removed it, and found that the wire had detached. Another X-ray was obtained which showed a foreign body on the right side, corresponding to the first lost wire (Figure A, solid box), and a second wire in the midline, quite distant from where the parents had initially inserted it (Figure A, dotted box).\nAt the time of surgery, the first wire was easily palpable within the subcutaneous tissue of the right lower quadrant abdominal wall and was removed via a small skin incision (Figure E). The second wire was not palpable, and an X-ray taken on the operating room table showed that the wire had migrated from the midline to the left upper quadrant (Figure B, dotted box).\nHaving concluded from these images that the second wire was likely within the peritoneal cavity, we performed a diagnostic laparoscopy. We were unable to locate the wire with direct inspection. With the aid of intraoperative fluoroscopy, the wire was ultimately found to be embedded within the omental tissue (Figure C and 1D). The piece of omentum encasing the wire was removed with electrocautery. The specimen was inspected grossly, and the wire was identified (Figure F). A postretrieval X-ray demonstrated successful removal of both wires.\nFinally, under direct laparoscopic visualization, we placed a new sensor percutaneously per instruction. The needle did indeed penetrate the peritoneum on initial insertion (Figure A). We then adjusted the angle of deployment of the needle so that we could place it without violating the peritoneum (Figure B). The findings were shared with the parents, our colleagues in endocrinology, and the manufacturer of the device. The patient was admitted overnight for monitoring and discharged the next day. He is currently using the same continuous glucose monitor system to help him maintain glycemic control.
A 54-year-old male with past medical history of end-stage renal disease on hemodialysis, hepatitis C, ischemic stroke, depression, and anxiety was brought to the emergency department with the complaints of fever (100.2°F) and increased surgical site pain in left thigh. He underwent creation of a left superficial femoral artery to greater saphenous vein prosthetic AV graft 1 day prior to admission. Few hours after the procedure, he started having increased surgical site pain, erythema, and small vesicles at the margins of wound. On examination, the left thigh AV fistula site staples were intact. There was mild erythema, tenderness, and vesicles at margins. Serous discharge was noted from vesicles but no purulent drainage from wound area. Lab investigations showed creatinine level of 12.6 mg/dL and a potassium level of 6.4 mEq/L at the time of admission prompting hemodialysis. On admission, his vitals were normal and white blood cell (WBC) count was 10 000/cumm. The next day, his left thigh wound pain and swelling worsened concomitant with erythema, tenderness, and purulent discharge. Blood cultures were taken and drainage sample was sent for culture and sensitivity. His WBC count increased to 13 000 and erythrocyte sedimentation rate was 45; therefore, on suspicion of AV graft infection he was started on vancomycin 1.25 g intravenously given after each hemodialysis session. The vancomycin trough level was therapeutic, and blood cultures and drainage culture were negative. Despite the antibiotic therapy, his symptoms worsened with each passing day. Repeat blood and drainage cultures were consistently negative. Computed tomography scan of pelvis showed extensive edema and congestion of the subcutaneous fat extending from the left external oblique musculature inferiorly along the lateral aspect of the pelvis into the left upper extremity (). Because of worsening symptoms, he underwent exploration of left thigh AV graft and samples were collected from perigraft tissue and perigraft fluid for culture and sensitivity. Once again, all cultures came back negative. Over the next couple of days, he started having purulent drainage from multiple locations along the skin overlying the graft (). The decision was then taken to surgically remove the graft. The drainage sample from excised graft was also negative for bacteria. After graft removal, his symptoms markedly improved with complete resolution of symptoms in 7 days. In the meantime, he was given topical emollients for symptom relief only. Due to abrupt onset of symptoms within hours after AV graft placement, the diagnosis of AV graft contact dermatitis was made.
A 75-year-old female had the habit of taking vinegar daily for 50 years. She was transported to our emergency room complaining of vomiting and loss of consciousness. Her medical history included hypertension, and she took amlodipine 10 mg daily. Her family history was negative. Socially, she denied any history of smoking, drug or alcohol use.\nOn physical examination, she was unconscious, hypotensive and in respiratory failure. Her blood pressure was 86/58 mmHg. An electrocardiogram showed sinus tachycardia. Oxygen saturation was 90% (10 L of oxygen administration). No subcutaneous emphysema was observed in the chest. There were no signs of peritoneal irritation. Laboratory data showed a white blood count of 3500. The C-reactive protein level was 8.7 mg/dL, and renal dysfunction was present. Electrolytes, liver function and urinalysis were all within normal ranges.\nComputed tomography of the thoracic and abdomen with contrast showed poor contrast in the middle intrathoracic esophagus, mediastinal emphysema, right pneumothorax and pleural effusion ().\nShe was diagnosed with septic shock due to esophageal rupture; therefore, we performed emergency surgery. We opened the chest through a posterolateral incision of the sixth intercostal space in the left-sided supine position. The right chest cavity was highly contaminated with fluid containing food residue. We identified a 25 mm perforation site in the middle intrathoracic esophagus. The esophageal mucosa turned black (). We determined that suture closure was not possible and resected the thoracic esophagus by 10 cm. She needed to have an esophageal fistula and an enteric fistula, but because her shock vitals were prolonged intraoperatively, she needed immediate systemic management in the intensive care unit, and the surgery was completed after esophagectomy and intrathoracic lavage drainage as a damage control strategy.\nIn the excised specimen, the esophageal mucosa was blackened throughout and there were no obvious malignant findings ().\nHistopathological examination revealed that the esophageal mucosa was ulcerated and necrotic and perforated in one place. Lymphocytic infiltration was observed in the mucosal surface layer; however, no findings were found beyond the muscle layer. The cause of perforation could not be determined ().\nIntensive care was started immediately after surgery, and, her vitals were stable 15 hours postoperatively. Therefore, we performed esophageal and enteric fistulae. The esophageal mucosa, which was an esophageal fistula, was also blackened (). Esophageal fistula mucosa normalized on the seventh postoperative day. After waiting for sufficient improvement in her general condition, she underwent esophageal reconstruction by the third operation 60 days after the first operation. We performed a posterior sternal pathway cervical esophagogastric anastomosis and cholecystectomy. She started receiving oral intake on the 12th day and was discharged on the 27th day after the third surgery.
A 6-year-old boy with no pathological history accidentally fell from the top of an approximately 3 m climbing pole and injured his right extended elbow and wrist joint. Due to pain and deformity in the right elbow and wrist joints, he visited our hospital. Swelling and a dinner fork deformity of the right wrist joint and pronounced swelling of the right elbow joint were observed. No skin damage was observed. No findings of nerve injury or arterial injury were obtained in the right upper limb. Radiography revealed lateral dislocation of the radial head, a fracture of the proximal ulnar metaphysis, and mild bending deformation at the fracture site. In addition, fractures of the distal radius and ulna, as well as dorsal displacement of the distal fragment, were seen (). Thus, the patient was diagnosed with Bado type III Monteggia injury with ipsilateral fracture of the distal radius and ulna.\nManual reduction under nerve block was attempted on the day of injury. However, because it was difficult to maintain the reduction of the radial head, as shown in , open reduction and percutaneous procedures were performed under general anesthesia. A Kirschner wire was inserted, percutaneously, from the olecranon into the ulnar diaphysis. When the Kirschner wire was in place, the dislocation of the radial head immediately showed good reduction. Further, open reduction and fixation of the fractured distal radius and ulna were performed with Kirschner wires (). A long-arm cast was used for external fixation with the elbow in 90° flexion and the forearm in an intermediate position.\nTwo weeks after surgery, callus formation at the fractured bone was observed. Therefore, the cast was removed, and range of motion (ROM) exercises of the elbow and wrist joints were initiated. Since bone union was achieved at 6 weeks postsurgery, the Kirschner wires were removed. Pain, ROM limitation, and lateral instability were not observed in the elbow or wrist joints at 3 months after surgery. Additionally, plain radiographs taken at the same time showed a radially convex curvature at the proximal portion of the ulna and lateral subluxation of the radial head (). However, a gradual correction in the outward displacement of the radial head was observed during the 3-year follow-up.\nTwenty-one years after surgery, the patient returned to our hospital for another disorder. At that time, we obtained informed consent to perform an examination and take radiographs of the previous Monteggia injury. Neither spontaneous pain, pain during exercise, tenderness, nor ROM asymmetry were observed (). The biocompatibility of the radiocapitellar joint was good, and no malunion was found in the distal radius and ulna (). The patient reports that he has been working as a computer programmer and performs weight training as a hobby without limitations.
A 44-year-old previously healthy Asian female reported to the neurology clinic with complaints of episodic, persistent, uncomfortable needle-like sensations in her genitalia extending to the anal area and the tip of the coccyx. She had been suffering from these symptoms for approximately 11 years. She had difficulty describing the actual nature of this phenomenon, which, according to her, was more of an irritation and discomfort than actual pain. The discomfort was aggravated during rest and periods of inactivity, particularly at night. It responded briefly to mefenamic acid tablets for four to five hours. She also reported the pain was worse before her monthly menstrual cycle but that sexual activity did not affect her discomfort. Sometimes, the discomfort was intense and would wake her from sleep causing severe discomfort and resulting in difficulty sitting; walking would relieve her symptoms partially. She was better in the morning, but the symptoms appeared again at end of the day. She had multiple gynecology and dermatology consults with no relief or clear diagnosis. She was prescribed antifungal creams, including fluconazole and ketoconazole, topical steroids (betamethasone and hydrocortisone), and mefenamic acid for pain relief. All failed to bring relief. She also consulted a homeopathy practitioner, who labeled her as suffering from a chronic skin disease and prescribed oral and topical medication. She took it for a few months but stopped it due to lack of relief. Ultimately, due to the distress and anguish associated with the condition, lack of response to various treatments, and social embarrassment, she stopped visiting doctors for some time. However, during a recent visit, the consulting gynecologist suspected it was not a dermatological or gynecological problem and referred her for a neurology consult.\nThere was no history of back pain, sensory symptoms in the legs, urinary incontinence, or neurological disorder, such as Parkinsonism or restless leg disorder. She had a stable and happy marital life of 22 years and had three children. She has used no substance of abuse or recreational drugs. Her neurological and gynecological examinations, as well as magnetic resonance imaging (MRI) of the spine, were normal (Figure ). Similarly, her biochemical profile (including complete blood count, urine analysis, serum urea, and creatinine) and an ultrasound examination of abdomen and pelvis were normal.\nConsidering her history of circadian rhythm variations in symptoms and aggravation on rest, RGS was considered the most likely cause of her symptoms. After a detailed discussion with the patient, she was prescribed 0.5 mg of ropinirole once at night. It was increased to twice daily after seven days, to which she responded well and reported a dramatic relief of symptoms after 14 days. The dose was increased to 1 mg in the morning and 0.5 mg at night, which led to further improvement. On her third follow-up after two months, she reported taking her medication regularly, which led to almost no episodes of discomfort, with exception of one or two times. The dose of ropinirole was maintained for one month. At the three-month follow-up, she reported only one episode of pain and itching, when she missed one dose of her medicine for a few days; otherwise, she had no complaints and reported no side effects of the medication.
A-82-year-old otherwise healthy male had a history of fever of 1 week. He was not a diabetic or hypertensive. There was no history of trauma. After few days, he developed headache with vomiting which was associated with low backache. This was followed with gradual onset of weakness of lower limbs and urinary incontinence. The weakness worsened over the next few days to flaccid paralysis in both lower limbs. He was taken to a local hospital from where he was referred to us. On examination, he was conscious but not oriented. There were patchy ecchymotic patches on his sacral region. GCS was E4V4M5 along with Grade 0 power of lower limbs. There was no perception of any sensations below the trunk. There was normal power in the upper limbs. Investigations revealed he had severe thrombocytopenia (platelet count 20,000/mm3) which prompted further evaluation for dengue in view of ongoing epidemic. He was positive for dengue IgM antibodies. His INR was 1.3 and APTT was 45 s. Emergency CT scan of the brain along with magnetic resonance imaging (MRI) of the spine was advised. CT scan revealed diffuse patchy subarachnoid hemorrhages involving both cerebral hemispheres along with intraventricular bleed [Figure and ]. MRI spine showed he had subdural and intramedullary hematoma involving the thoracolumbar region. The spinal cord was distended from T11 to L1 regions due to underlying hematoma [Figures and ]. It was decided to perform emergency decompression of the spinal cord, but his platelet counts were too low to perform a safe surgery. He was given multiple platelet transfusions after which thrombocytopenia slowly recovered partially to more than one lac over a period of 4 days after admission. He was taken up for surgery during which, a laminectomy of the T11, T12, and L1 vertebrae was performed. The cord was seen swollen with underlying bluish blush of the hematoma []. The dura was opened to visualize the underlying subdural clot which was removed slowly with biopsy forceps []. The hematoma was seen to enter the substance of the spinal cord and was splaying the nerve roots. Thorough irrigation with saline was given and the hematoma was evacuated in piecemeal. At the end of the surgery, the cord was seen pulsating after which a meticulous dural closure was achieved. He regained consciousness after surgery but the power in his lower limbs did not improve.
A 60-year-old Caucasian woman was admitted to our hospital because of severe upper gastrointestinal bleeding. An endoscopy was performed, revealing bleeding from gastric varices in the subcardial region. Due to the large variceal size, endoscopic therapy with variceal ligation could not be performed. She was transferred to our intensive care unit (ICU).\nShe had a history of similar episode of massive gastrointestinal bleeding from gastric varices six years previously. Evaluation at that time with liver function tests, portal venous flow and magnetic resonance angiography did not reveal an identifiable cause.\nOn admission to the ICU our patient initially showed no signs of hypovolemic shock. Laboratory results again did not indicate impaired liver function. Esophagogastroduodenoscopy again revealed bleeding from subcardial gastric varices in the absence of evidence of esophageal varices (Figure ).\nEndoscopic treatment with histoacryl glue only resulted in a temporary reduction of the bleeding. To reduce the portal venous pressure the vasopressin analog terlipressin was administered. Despite this therapy, there was another severe episode of upper gastrointestinal bleeding with signs of shock. In all, 10 units of red blood cell concentrate and four units of fresh frozen plasma were transfused. Sufficient endoscopic therapy could not be achieved. An abdominal sonograph showed she had an enlarged spleen (15.9 × 5.4 cm; liver size and structure were normal, with normal flow in the portal vein). A computed tomography (CT) scan showed total occlusion of the splenic vein. Despite the limited data on urgent splenic artery embolization, she was subsequently referred for interventional radiological procedures. A successful splenic artery embolization was performed via the transcatheter approach, depositing coils into the splenic artery resulting in immediate cessation of variceal bleeding (Figure ).\nNo recurrence of bleeding was noted post-embolization. After embolization, our patient complained of mild left upper abdominal discomfort, which was effectively relieved by routine analgesics.\nAt two weeks after admission our patient was fully recovered and was released from our department. There were no further bleeding complications for 18 months. Follow-up endoscopy was performed two months and eight months after the intervention, showing only mild gastric varices without signs of bleeding.
A 35-year-old man sustained a blunt traumatic injury to the pancreas with pancreatic duct rupture in the head of the pancreas (). He was initially treated by operative drainage of the leaking pancreatic fluid and insertion of a drain into the dilated distal pancreatic duct (). Two years later, the duct drain prolapsed and was difficult to reinsert. The patient developed abdominal and low back pain; therefore, we created a side-to-side anastomosis between the dilated distal pancreatic duct and jejunum as a Roux-en-Y limb ().\nEighteen months after the second operation, the patient developed chronic abdominal pain and gastrointestinal bleeding from the Roux limb. He exhibited similar symptoms several times, and nonoperative management with endoscopic hemostasis was successful each time. The remainder of the patient’s history was unremarkable.\nThe patient presented to our institution at 48 years of age with worsening abdominal pain and recurrent gastrointestinal bleeding. The physical examination was unremarkable. Laboratory studies showed a decrease in the hemoglobin concentration from 12.3 to 9.4 g/dL over 1 day and a slight increase in the blood urea nitrogen concentration to 27 mg/dL. Extravasation of intravenous contrast was not seen on an enhanced computed tomography scan. Double-balloon endoscopy showed multiple erosions and ulcers in the Roux limb with bloody enteric content and fresh blood (). The patient was diagnosed with anemia due to gastrointestinal bleeding from the Roux limb; however, a specific site of bleeding could not be identified. In spite of transfusion of 4 units of red cell concentrate after hospitalization, the bleeding continued and the anemia progressed to a hemoglobin concentration of 7.0 g/dL. Therefore, we decided to perform emergency surgery.\nThe patient underwent resection of the Roux limb, which was the source of bleeding, and central pancreatectomy including the area of the anastomosis and extending to the main pancreatic duct. Because both the head and tail of the pancreatic parenchyma appeared normal, including the injured pancreas head, we attempted to preserve both parts of the remaining pancreas. We considered the distal pancreatic fluid outflow tract from the pancreatic head and performed primary reconstruction with a double pancreaticogastrostomy without using the jejunum to avoid recurrent gastrointestinal bleeding. An incision was made in the anterior wall of the stomach, and the pancreaticogastrostomy was performed such that the stump of the pancreatic head was invaginated into the posterior wall at the gastric antrum, and the stump of the pancreatic tail was invaginated into the posterior wall of the proximal stomach (). A drain was inserted into each pancreatic duct to create an incomplete external fistula through the anterior wall of the stomach. The patient was discharged 34 days postoperatively and developed a Grade B pancreatic fistula (International Study Group of Pancreatic Surgery classification). Although the drain amylase concentration was high and we had to maintain drainage for more than 3 weeks postoperatively, no fluid collection or cavity was present in the abdomen. We treated the pancreatic fistula by exchanging the drain two times. The drain in the duct of the pancreatic tail spontaneously fell out. The drain in the duct of the pancreatic head remained in place and was transected and internalized endoscopically before discharge.\nThe surgical specimen showed multiple nonspecific erosions and ulcers in the resected Roux limb (). The patient was followed up every 3 months and had not developed diabetes or any nutritional disturbances by 1 year postoperatively. The remaining pancreatic tissue, including the distal pancreatic head, appeared normal without atrophy on a computed tomography scan. He was doing well without abdominal pain or evidence of gastrointestinal bleeding at the time of this writing.
An 84-year-old female patient was admitted to the accident and emergency (AE) department of our hospital complaining of painful restriction of movement in the left hip for the past month. She had undergone a left cement less THA (reflection cup, synergy stem, 28 mm ceramic on XLPE bearing coupling, Smith and nephew, Memphis, Tennessee, USA), through an ordinary posterior approach, 14 years previously. The immediate post-operative period had been uneventful. In the 8th post-operative year, during a follow-up evaluation, the patient was found to be asymptomatic, but signs of PE wear were recorded on radiological evaluation. The patient was advised, by her surgeon, to attend outpatient clinics at regular intervals and she was informed about the possibility of an early revision of the bearing surface. Despite this, she failed to attend any other appointments. On admission to the AE, the patient reported that she had experienced increasing restriction of movement and functional impairment during the previous year without having sustained a recent hip injury. On clinical examination, painful restriction of movement, no signs of soft tissue injury or infection, and a leg length discrepancy of 4 cm were recorded. Laboratory blood workout was found to be normal. On radiological examination, a left THA dislocation was found () and the patient underwent three unsuccessful attempts at closed reduction, under anesthesia, in the AE department. During these attempts, the patient sustained an undisplaced fracture of the neck of ipsilateral fibular head. The following day, during an open reduction under anesthesia in the operating theater, a central dislocation of the head of the prosthesis was found. The PE liner was found to be worn and broken, and the ceramic head had centrally perforated the metallic acetabular shell and was located behind the shell in an osteolytic area full of PE and metallic debris. Excessive soft tissue metallosis was also found. The femoral stem and the perforated metallic shell were found to be stable. A thorough soft tissue debridement was performed, the retro-acetabular bone defect was filled with cement and a PE liner was cemented into the damaged metallic shell (). The prosthesis was then reduced and evaluated for stability using a 32 mm ceramic head. The patient experienced an uneventful post-operative clinical and functional recovery.
An otherwise healthy 19-year-old male soccer player experienced left ankle pain when playing soccer at a competitive university level. The pain was severe enough that he could not continue with practice, so he consulted a local doctor the following day. A plain radiograph and three-dimensional computed tomography scan demonstrated osteophytes at the anterior edge of the distal end of the tibia and dorsal talar neck, indicating anterior ankle impingement syndrome (Figures and ). The patient was referred to our department 2 weeks later. On physical examination, there was tenderness over the anteromedial aspect of the ankle and range of motion at the ankle joint was limited to 5° of dorsiflexion.\nThree weeks after the first presentation to us, he underwent arthroscopic debridement of anterior tibiotalar osteophytes and synovectomy for the anterior ankle impingement syndrome. Under general anesthesia and with a tourniquet, noninvasive distraction was applied using an ankle strap and foot traction. An arthroscopic pump was used to distend the joint. After making an initial incision through the skin, a mosquito hemostat was used to introduce the arthroscope into the articular cavity and create standard anteromedial and anterolateral portals. Ankle arthroscopy revealed large osteophytes at the anterior edge of the distal end of the tibia and the dorsal talar neck (), which were excised using a bone cutter (). The hypertrophic synovium was resected arthroscopically using a 3.5 mm motorized shaver. On further inspection of the ankle, the cartilage was seen to be normal. The joint was irrigated thoroughly and the portal sites were closed with nylon. Resection of the osteophytes was confirmed after ankle arthroscopy by plain radiography and three-dimensional computed tomography (Figures and ). The patient was permitted to bear weight on his left foot as tolerable as possible\nThe patient experienced no pain in the early postoperative period. After ankle arthroscopy, a normal dorsal artery pulse was palpable. Three days after the ankle arthroscopy, the patient twisted his left leg accidentally, such that his full body weight was suddenly supported by the left foot. Later that day, the patient presented with pain and swelling in the anterior aspect of the ankle (), which he considered to be bearable. The sutures were removed 10 days after the ankle arthroscopy.\nTwelve days after the arthroscopy, magnetic resonance imaging showed a 25 × 22 × 13 mm pseudoaneurysm with a heterogeneous low-intensity to isointensity signal on T2-weighted imaging () and a high-intensity signal on T2 star-weighted imaging at the level of the anterior ankle joint ().\nThirteen days after arthroscopy, color and duplex Doppler ultrasonographic examination was performed by the radiology team at our hospital. It showed a mosaic pattern of colors with the “whirling blood flow” and “to-and-fro” motion typical of a pseudoaneurysm on the posterior wall of the distal portion of the anterior tibial artery (). The final diagnosis was pseudoaneurysm caused by injury of the anterior tibial artery during ankle arthroscopy. The patient was then referred to the cardiovascular surgery team at our hospital. Catheter angiography of the lower limb vasculature was performed to obtain detailed information on the state and integrity of the anterior tibial artery and determine the direction of flow.\nFourteen days after arthroscopy, angiography was performed by the radiology team at our hospital. It revealed a pseudoaneurysm with an intact posterior tibial artery and plantar arch circulation (). Nineteen days after arthroscopy, an anastomosis was created for the injured anterior tibial artery to prevent progression of the ankle pain and swelling. A transverse incision was made over the anterior aspect of the ankle. The pseudoaneurysm was believed to be caused by disruption of the posterior 1/3 of the anterior tibial artery wall () and leakage of blood into the ankle, forming a pseudoaneurysm. The anterior tibial artery was isolated and clipped proximally and distally. The injured arterial walls facing each other were sutured longitudinally with preservation of blood flow distally, although the diameter of the artery became narrower at this point ().\nApart from slight paresthesia of the first web space, the patient's subsequent postoperative course was uneventful and he was discharged from hospital 1 week later. The ankle pain and swelling had resolved immediately after repair of the anterior tibial artery injury. He returned to playing soccer at a competitive university level 4 months later.
The patient was a 64-year-old man with destructive injury of both lower extremities due to a machine accident. His left ankle and heel bone, along with the soft tissue, were torn off, and the peripheral blood supply and sensation to the toes of the left foot were lost. We amputated his left leg below the knee. The left fibular head was retained after the left foot operation. Two months later, the patient came to our hospital for additional treatment after debridement of necrotic tissue operations twice. There was still approximately a 20 ×10-cm area on the right foot and right external ankle exposing the distal fibula fractures. X-ray showed that the right external ankle bony defect and the right medial ankle mortise widened; fortunately, the left fibular head was retained after left foot amputation (Fig. ). We reconstructed the right ankle using vascular anastomosis of the fibular head and a flap taken from the left stump. The Ethics Committee of the Second Affiliated Hospital of Soochow University approved the study.\nWe used an ultrasonic Doppler instrument (Model: ES-1000SPM Hayashi Denki Co., Ltd, Hong Kong China) to find the perforating branch of the peroneal artery before surgery, and near that location we created a flap approximately 20 × 10 cm in size according to the right foot wound (Fig. ). Surgery was performed with the patient under general anesthesia. The patient was placed in the supine position on the operating table with the left knee joint slightly bent and the left lower limb stump in internal torsion. The incision began in the popliteal space, swept down to the fibular head, and continued along the lateral peroneus muscles to the stump extremity. We cut the skin and subcutaneous tissue, then separated the peroneus longus and soleus muscles. We found the nervus peroneus communis on the inner posterior margin of the biceps femoris tendon and dissociated it for protection. The peroneal perforator was located in the intermuscular space between the peroneus longus and triceps surae muscles, then we freed the peroneal artery and 2 accompanying veins. We cut off the biceps femoris tendon, fibular collateral ligament and other muscle tissue attached to the fibula, extracted the fibular head, and then divided the superior tibiofibular joint. On release of the tourniquet, bleeding spots on the fibula and residual muscle tissue showed that the blood supply was satisfied. Therefore, we could then cut off the vessels and take the fibular head (Fig. ). We sewed the biceps femoris tendon and fibular collateral ligament with deep fascia around the lateral tibial plateau to avoid instability of the knee joint. We completely debrided bone splinters and necrotic soft tissue from the lower right limb and smoothed the edges. The free fibular head was fixated to the distal fibula with a fibular plate. The reconstructed inferior tibiofibular joint was reinforced with 3 lag screws before anastomosis of the vessels in the transplanted fibular head with the recipient area. Lastly, we made a suture flap in the wound and inserted rubber drainages (Fig. ).\nDue to necrosis of the skin and soft tissue of the distal flap 7 days after the operation, part of the reconstructed external ankle bone was exposed, so we transferred the sural neurocutaneous vascular flap to repair the skin and soft tissue defects of the lateral malleolus. X-ray examination 3 months after the operation showed that the fracture had a bony union and the shape of the transplanted fibular head was similar to the normal external ankle. At 12-month follow-up, walking function had been recovered with the help of a left artificial limb (Fig. ). According to the Baird–Jackson scoring system, the curative effect was satisfied.
A 28-year-old male presents with a past medical history significant for cerebral palsy, fully dependent in activities of daily living. He has had a lifelong history of intermittent abdominal distension and constipation. He had previously presented to an outside hospital due to low-grade fever with intractable nausea and vomiting. A computed tomography (CT) abdomen pelvis with contrast demonstrated findings consistent with pseudoobstruction vs. ileus, possibly due to cecal bascule or volvulus. Due to failure to improve with nonoperative measures, he underwent a decompressive colonoscopy to reduce the colonic distension. He was subsequently taken to the operating room where a cecal bascule was identified, per outside operative record, as well as a severely dilated small bowel and redundant colon. He underwent a diverting loop ileostomy, gastrostomy tube placement, and appendectomy. Reasons for this operative decision-making are not made known to the authors. His postoperative course was complicated by delayed return of bowel function requiring total parenteral nutrition.\nApproximately 6 weeks later, he presented to our emergency department with fever, leukocytosis, abdominal discomfort, and multiple episodes of emesis. He remained hemodynamically stable, but his stoma had prolapsed at least 25-30 cm and appeared edematous and dark red distally (). He was taken to the operating room where further examination of the stoma demonstrated approximately 40 cm of prolapse. The mucocutaneous junction of the prolapsed portion was dissected to delineate anatomy (). The mucosa was noted to be edematous and abnormal. At this point, it became apparent that the prolapsed portion was the efferent limb of the loop ileostomy with the intussuscepted cecum and the entire right colon. The decision was made to proceed with a right hemicolectomy and mucous fistula creation at the level of the transverse colon to eliminate the mobile bowel (Figures and ). This entire resection was done through the stoma site. Primary anastomosis was not performed due to the patient's poor nutritional status and need for stimulation to have a bowel movement due to spasticity. An end ileostomy and transverse colonic mucous fistula were created through the previous stoma site. The patient's postoperative course was complicated by delayed return of bowel function. He was eventually discharged home with his family on postoperative day 9 tolerating tube feeding through his gastrostomy tube with adequate ileostomy function. He was seen in the clinic postoperatively and recovered to his baseline.
The second patient, the 75-year-old man with severe depression (see under results Case 2), experienced a prolonged seizure, which was stopped by intravenous application of benzodiazepine. It is common sense among psychiatrists to limit ECT-induced seizure; however, there is a debate on after which duration a prolonged seizure should be terminated.\nFears of prolonged seizures induced by ECT are manyfold, which are found in doctors more than patients. While the fear to end up in a status epilepticus ranks high, it is reported to occur mainly for single cases (; ; ). Prolonged seizure is found more often to be 1–2% in the clinical routine where termination by medication runs smoothly (). Analyses of ECTs in clinical routine found that women have longer seizures but only at the first treatment session, a strong inverse correlation between age and seizure duration, and a drop in seizure duration along a course of treatments (; ).\nGiven that many psychotropic medications are thought to either promote or prevent seizures, there is an ongoing concern about concurrent psychotropic medication and ECT administration. In cohort studies, benzodiazepines, antiepileptic agents, selective serotonin-reuptake inhibitors, tricyclic and tetracyclic antidepressants, and stimulants were not associated with seizure threshold or duration, indicating that psychotropic medications may have little effect on seizure at ECT initiation (). Bupropion showed contradictory results of unimpaired () or reduced seizure duration () but also to cause partial status epilepticus (). Several other medications increase the duration of seizures, such as caffeine (), antibiotics (), or remifentanil (). There was no uncontrollable risk with these substances.\nIn clinical practice, suddenly occurring prolonged seizures can never be ruled out particularly in treatment-resistant patients with combinations of psychotropic drugs (). This, however, could be ruled out widely for our patients who received moderate doses of mirtazapine and duloxetine only. His longer seizure length and several hours period to restore normal consciousness is typically more often occurring with high stimulus intensity. High stimulus intensity is among one of the other factors associated with prolonged reorientation time following seizure as well as an increased risk of post-ECT delirium (). Benzodiazepine application was necessary to stop the seizure, and it should be considered that benzodiazepines such as lorazepam have been shown to induce prolonged apnea after ECT-induced prolonged seizure. This negative respiratory effect appears to be rare () and occurs primarily in persons who are acetylcholine (ACH)-deficient and exhibit increased succinylcholine efficacy as described earlier.\nThe take-home message is that medical termination will be required for patients who could always exhibit a prolonged seizure induced by ECT, which may induce prolonged apnea, and clinicians should be prepared for this contingency.
We present a 29-year-old Asian woman who underwent fertility-sparing surgery at 15 weeks of pregnancy for ovarian cancer, later undergoing emergent cesarean section and exploratory laparotomy at 31 weeks of pregnancy for recurrence of ovarian cancer. She has provided written permission for the case to be reported. There was neither family history of cancer nor past medical history.\nThe patient, gravida 2, para 0, presented to her local obstetrician complaining of abdominal pain. On gynecologic examination, a 9 cm sized ovarian cyst was found by ultrasound examination. For surgical intervention, she was referred to a nearby hospital, where intrauterine pregnancy as well as ovarian cyst was found. Thus, she was referred to our hospital for the management of pregnancy complicated with an ovarian cyst. At the initial examination, sonographic evaluation suggested no malignancy: there was no ascites, Doppler ultrasound revealed no increased blood flow, and grayscale patterns appeared normal. Pelvic magnetic resonance imaging (MRI) examination also showed the possibility of benign ovarian cyst (). The tumor markers were within normal limits (CA125, 27 U/mL; CA19-9, 17 U/mL). Considering the possibility of benign ovarian cyst, we continued regular prenatal checkup, setting surgical treatment at 15 weeks of pregnancy. In her first trimester of pregnancy, she had recurrent episodes of mild abdominal pain and vaginal bleeding, warranting admission.\nAt 14 weeks and 0 days of gestation, the patient suddenly complained of severe left abdominal pain. On suspicion of torsion, the patient underwent emergent laparotomy. During surgery, 10 cm × 8 cm multiple left ovarian cysts and one rotation of the cyst were noted. With a diagnosis of torsion of ovarian cyst, we performed cystectomy of the left ovarian tumor. As a 1 cm sized small nodule on the wall of the specimen was noted, we performed careful exploration and washing cytology. The right ovary, uterus, and other intra-abdominal regions were normal. The postoperative course was uneventful. The final pathology was consistent with high-grade mucinous adenocarcinoma, endocervical type (Figures and ). Washing cytology was negative. After a discussion involving the patient and her family, gynecologic oncologist, and maternal-fetal medicine specialist, secondary laparotomy as fertility-sparing surgery was selected.\nAt 15 weeks and 2 days of gestation, the patient underwent fertility-sparing surgery: exploratory laparotomy, left salpingo-oophorectomy, infracolic omentectomy, and cytologic examination of ascitic fluid. Intraoperative exploration showed a small amount of ascites and no residual lesion on the diaphragm, the surface of the liver, and Douglas' pouch. Lymph nodes were not palpated in the pelvis and para-aortic lesion. The postoperative course was uneventful except for postoperative ileus, treated with fasting and intravenous infusion from postoperative days 6-9. Final pathology showed no residual lesion on the omentum, and washing cytology was negative. A small atypical cell with large nuclei was found in the left residual ovary. Considering the fact that tumor was detected on the surface of the left residual ovary, the primary tumor stage was presumed to be ovarian cancer (FIGO stage IC2). After a repeated discussion, we omitted chemotherapy during pregnancy and continued the prenatal checkups in the outpatient department every 2 weeks.\nAt 31 weeks and 0 days of gestation, she called our department and complained of rapidly increasing abdominal circumference and nausea. Regular prenatal checkup findings 9 days before, including transvaginal ultrasound, were normal. Upon arrival, the patient appeared sick and ultrasound revealed massive ascites. Due to increasing dyspnea, paracentesis (approximately 3 L of ascitic fluid) was performed. Cytologic examination of ascetic fluid showed malignant cells. CA125 and CA19-9 levels were elevated by up to 317 U/mL and 3656 U/mL, respectively. Sooner after admission, oliguria/anuria developed acutely, which was caused by massive ascites and intravascular dehydration. Corticosteroids (two intramuscular injections of betamethasone of 12 mg 24 hours apart) were started to be administered for fetal lung maturity.\nAt 31 weeks and 3 days of gestation, blood urea nitrogen (BUN) and creatinine (Cr) were elevated due to uncontrolled anuria (BUN; 30.9 mg/dL, Cr: 1.12 mg/dL). Making a diagnosis of acute kidney injury, we performed emergent cesarean section. On surgical finding, there were a number of 1 cm sized nodules in the small bowel wall and <1 cm sized nodules in the abdominal wall and peritoneum. A total of 3.2 L of ascitic fluid was removed. There was no gross residual disease on the right ovary and appendix. The placenta and umbilical cord were normal, grossly and pathologically. Intraoperative biopsy confirmed metastatic ovarian carcinoma.\nThe infant was male and weighed 1422 g with an Apgar score of 8 and 9 at 1 and 5 min, respectively. The fetal blood counts were normal at the time of delivery. The infant showed minor acute respiratory distress syndrome and mild anemia but had no neurologic, psychomotor, or developmental abnormalities and was discharged 60 days after birth.\nAfter cesarean section, the patient was transferred to the intensive care unit. Oliguria continued due to rapid accumulation of ascites in the early postpartum period. For management of malignant massive ascites, we repeated cell-free and concentrated ascites reinfusion therapy and maintained the amount of urine output. The baseline renal function tests improved gradually to the limit of normal on postoperative day 5. On postoperative 10 days, adjuvant chemotherapy consisting of paclitaxel (135 mg/m2) and carboplatin (the area under the curve: AUC, 5) was performed. In this period, the general condition of the patient was not good due to the massive ascites and anorexia. So, we chose the lower dosage of paclitaxel. Toxicity was evaluated according to the National Cancer Institute common toxicity criteria. The patient developed grade 3 neutropenia as a side effect. Meanwhile, after the first cycle of chemotherapy, she had pain and anorexia caused by massive ascites and abdominal bloating, which was controlled with total parenteral nutrition and continuous opioid infusion. After two cycles of chemotherapy, ascites decreased gradually and opioids were discontinued. Chemotherapy was repeated every 3 weeks, according to standard protocols. Two weeks after the first cycle of carboplatin/paclitaxel, tumor markers had increased to a peak value (CA125, 311 U/mL; CA19-9, 6095 U/mL). Thereafter, the markers gradually decreased. She tolerated the chemotherapy well and was discharged after three courses of chemotherapy. However, after six courses of chemotherapy, she suddenly complained of nausea and anorexia. CT imaging showed cancerous ileus and ascites fluids. After the discussion with the medical team, the patient chose palliative care.
A 55-year-old Caucasian woman with a 5-month history of abdominal pain and vomiting was diagnosed as having a probable high-grade ovarian malignancy with a large volume of peritoneal disease. She had a prior ultrasound of her pelvis which demonstrated a 17 cm large irregular solid vascularized mass in her right ovary. Her comorbidities included obesity (body mass index of 33), asthma, and she had previously undergone a laparoscopic cholecystectomy. There was no significant family history. She did not smoke tobacco and she drank alcohol occasionally. She was scheduled for debulking surgery; however, she presented to our emergency department with worsening abdominal pain, vomiting, and diarrhea. A physical examination showed dry mucous membranes, capillary refill < 3 seconds, and jugular venous pressure of 4 cm. Her chest was clear on auscultation with dual heart sounds. Her abdomen was distended with generalized tenderness but no guarding or signs of peritonism. Bowel sounds were present. A computed tomography (CT) scan of her abdomen and pelvis was performed which demonstrated the large right ovarian tumor, peritoneal tumor deposits, and ascites. There was extrinsic compression of her sigmoid colon due to the tumor without radiological signs of large bowel obstruction. There were no other abnormalities of her bowel. She received intravenously administered fluids for rehydration, anti-emetics for nausea, and intravenously administered morphine for abdominal pain. She remained overnight in our emergency department for treatment. She was reassessed the following morning after resolution of her symptoms and was found to be hemodynamically stable and subsequently discharged home.\nShe re-presented 1 week later with similar symptoms and was admitted to hospital for further management. Following multidisciplinary discussion, she was recommended for neoadjuvant chemotherapy prior to surgical debulking. An urgent core biopsy of the mass confirmed the likely diagnosis of ovarian malignancy. This biopsy indicated a high-grade serous adenocarcinoma. The histology and CT findings were consistent with an International Federation of Gynaecology and Obstetrics (FIGO) stage III ovarian cancer. She was appropriately counselled as to the benefits and risks of chemotherapy prior to commencing treatment.\nShe was commenced on a first cycle of the commonly used platinum-based two-drug chemotherapy regime of paclitaxel and carboplatin []. The dose prescribed was a three weekly cycle of paclitaxel 100 mg/m2 and carboplatin 385 mg/body to achieve area under the curve (AUC) of 5 using the Calvert formula. The results of her pre-chemotherapy blood tests were within acceptable ranges. Specifically, her white cell count (WCC) was 10.1 × 109/L (normal 4–11 × 109/L) and neutrophils were 8.5 × 109/L (normal 1.5–8 × 109/L). She reported feeling better 1 day after chemotherapy. However, 3 days following the commencement of chemotherapy, persistent diarrhea developed. Stool cultures were negative for stool pathogens including Clostridium difficile. On the sixth day post-chemotherapy, she became febrile and acutely unwell with severe abdominal pain. Blood tests at the time revealed a WCC of 0.6 × 109/L and neutropenia of 0.1 × 109/L. A repeat CT of her abdomen and pelvis showed a thickened descending colon and rectal pneumatosis with perforation into the mesorectum (Figs. and ). Blood cultures were positive for Escherichia coli and meropenem was commenced. She was transferred to our intensive care unit for hemodynamic support due to septic shock. A surgical assessment was conducted but immediate surgical intervention was deemed inappropriate due to high predicted mortality. She was administered granulocyte colony-stimulating factor but her white blood cell counts did not improve and her respiratory function deteriorated. She died 9 days after the administration of the first dose of chemotherapy. An autopsy was not performed due to the wishes of our patient’s family.
A 41-year-old female, who was crushed from behind by a car while cycling, was admitted to our hospital with a bleeding wound in the medial of the right hip and the right lower limb unable to move for 12 h. Because her blood pressure was undetectable on admission to the local hospital, she received a transfusion of 6 units of red blood cells (RBCs). After ligation of the right external iliac artery and vein and wound suturing in the emergency room, the patient was transferred to our hospital. Physical examination indicated that the patient’s blood pressure was 138/70 mmHg and pulse rate was 120 per minute. She was conscious with a 40 cm long sutured wound, which extended from the right ilium, passed through the medial of the right hip and ended in the right side of the anus. The patient had no sensory and motor function in her right lower extremity, and had poor peripheral circulation and extensive soft-tissue injury in her right lower abdomen and right thigh. A diagnosis of hemorrhagic shock and traumatic hemipelvectomy of the right side was made. A compressive pelvic band was used to cover the wounds. Following active resuscitation rescue, pelvic angiography was performed 6 hours after admission. Angiography showed that the right external iliac and partial internal iliac arteries were transected and occluded by thrombi, and no obvious bleeding was observed. To reduce blood loss during the amputation, the left internal iliac artery was embolized. Because of the extensive soft tissue injury and injury of the iliac artery, salvage of the injured leg is impossible. Right hemipelvectomy was performed and the wound was closed 14 hours after admission. Because of severe wound infection, debridement was required every 3 or 4 days. A total of eleven debridement and skin grafting procedures were underwent. The patient’s wound was in stable condition, and she could walk with her crutches at a 12-month follow-up after the surgery (Figure \n).
A 67-year-old man underwent bioprosthetic aortic valve replacement and coronary artery bypass grafting with a saphenous vein graft to a diagonal artery in 2012 for severe aortic stenosis with coronary artery disease. He had done well until October 2016 when he presented with a five day history of fevers, chills and abdominal pain. On physical examination, a new 2/6 early-mid peaking systolic murmur was appreciated at the right upper sternal border. Six of six blood culture bottles were positive for Staphylococcus epidermidis and he was treated with vancomycin, gentamicin and rifampin for endocarditis while undergoing further evaluation.\nCT scan of the abdomen and pelvis noted a large splenic infarct consistent with a septic embolus. Transesophageal echocardiography demonstrated 1.8 × 0.9 cm2 vegetation on the prosthetic aortic valve with a large aortic root abscess. After confirming the diagnosis of endocarditis, surgery was scheduled for the next day given hemodynamic stability and the patients’ strong personal preference to delay surgery in order for family to be present despite understanding the risks of continued septic embolization.\nOn the morning of surgery, the patient complained of acute, severe chest discomfort and was diaphoretic and pale. EKG demonstrated new ST depressions in V1–V4 (Fig. ). CT coronary angiography was considered, however, given an evolving acute coronary syndrome (ACS), the decision was made to proceed for emergent coronary angiography, understanding the risks of possible iatrogenic embolization, but the benefit of rapid revascularization. The time to revascularization following lesion identification on CT and subsequent re-operative cardiac surgery was felt to be time prohibitive. Angiography revealed a 100% left circumflex ostial occlusion (Fig. ). The saphenous vein to the diagonal graft was widely patent and no additional lesions were identified. The presumed etiology of the new circumflex occlusion was a septic embolism secondary to prosthetic valve endocarditis. After discussions with the cardiac surgeon, referring cardiologist and patient, we felt that a hybrid strategy of attempted percutaneous coronary intervention with aspiration thrombectomy followed by open surgical valve replacement provided the optimal benefit/risk profile. Aspiration thombectomy was performed with an Export® AP thrombectomy catheter (Medtronic Inc., Minneapolis, MN) retrieving a substantial amount of organized material concerning for thrombo-vegetation (Fig. ). After two passes of the aspiration catheter TIMI 3 flow was restored (Fig. ), ST depressions resolved, and he experienced significant improvement in his chest pain.\nShortly after aspiration thrombectomy, the patient was brought to the operating room where he underwent re-do sternotomy, aortic root replacement utilizing 23 mm cryopreserved aortic homograft valve conduit as a free standing aortic root. The surgery was without complication.\nHis post-operative course was complicated by a drug reaction with eosinophilia and fever, likely related to either rifampin or vancomycin. Consequently, he was changed to intravenous daptomycin and was discharged to home on post-operative Day 19 with a 6-week course intravenous of antibiotics.
A 53-year-old female presented with gradually progressive diminution of vision in her left eye for 2 months. She had a history of diabetes mellitus for 20 years for which she is taking insulin. Her most recent HbA1C was 7.5%. She had no history of hypertension or renal problems. She had no past ocular history. Examination revealed a corrected distance visual acuity (CDVA) of 20/40 in her right eye and 20/100 in the left. Anterior segment examination showed nuclear sclerosis in both eyes. Posterior segment examination revealed intraretinal hemorrhages in all 4 quadrants indicating severe nonproliferative diabetic retinopathy in both eyes with clinically significant macular edema in the left eye confirmed by fluorescein angiography (). Spectral domain optical coherence tomography (OCT) of the macula was done and revealed multiple cystic spaces, mild subfoveal neurosensory detachment, and diffuse retinal thickening with a central subfield macular thickness of 332 μm in the left eye (). The right eye showed only few cystic spaces with minimal thickening. OCTA (Optovue, Inc., Fremont, CA, USA) was done in both eyes and showed areas of capillary nonperfusion in the superficial capillary plexus (SCP) of the maculae of both eyes (Figures and ). Three monthly intravitreal bevacizumab injections were done to treat the macular edema in the left eye. One month following the last intravitreal injection, CDVA improved to 20/60 in the left eye and was stable in the right eye. OCT showed improvement of the macular edema in the left eye (). OCTA was performed in both eyes and showed decreased vascular density of the SCP of the left eye compared to pretreatment OCTA while a mild increase was noted in the vascular density of the SCP of the untreated right eye (Figures and ). The patient was then followed up without requiring further intravitreal injections and 4 months following the last intravitreal injection OCTA was repeated in both eyes and showed improvement of the vascular density of the SCP of the left eye with unchanged SCP in the right eye (Figures and ). CDVA was 20/60 in the right eye and 20/100 in the left. Clinical examination revealed retinal neovascularization in the right eye with clinically significant macular edema in both eyes. OCT showed increased center-involving macular edema in both eyes with recurrent neurosensory detachment in the left eye (). Three monthly intravitreal bevacizumab injections were then done for treatment of proliferative diabetic retinopathy in the right eye and the macular edema in both eyes. One month following the last intravitreal injection, CDVA was 20/40 in the right eye and 20/60 in the left, OCT showed decreased macular thickness in both eyes (), and OCTA revealed decreased vascular density of the SCP of both eyes (Figures and ). Changes in the deep capillary plexus closely followed those in the SCP at all stages.
A 39-year-old woman of Indo-Trinidaian descent was referred for investigation of possible multiple sclerosis after periventricular white matter changes were noted on MRI of the brain following assessment for intermittent blurred vision of 8 weeks duration. At age 8, she had noticed bald spots on her head, which were followed at age 11 by three dark lines that appeared on her forehead and right side of her face. At 18 years of age, she had her first partial seizure and experienced occasional headaches and intermittent visual loss, which lasted for a few seconds followed by generalized tonic-clonic seizures, which was managed with carbamazepine 400 mg orally twice daily. She had approximately two to three seizures per year, which occurred only when she missed meals. At age 21, she noted upward deviation of her cheek-bones. After she had her first child at age 25, her chin deviated down and dark marks appeared at the middle of her chin and at the right side of her mouth. She had no episodes of status epilepticus. Despite seeing many doctors of diverse specialties over the intervening years the PRS/ECDS was not recognized due to unfamiliarity with the entity. At age 39, when she presented to us, she complained of blurred vision in the 2 preceding months, whenever she focused on obycts. Her ophthalmologist assessment reported anisocoria with the right pupil larger than the left (). Visual acuity was normal. In particular, there was no hypertension, diabetes, dyslipidemia, smoking, family history of intracranial, collagen vascular disease or stroke.\nShe went on to complete her secondary school education and then completed an Accounting degree. She had two children at the maternal age of 25 and 29 respectfully.\nThere was asymmetry of the right half of the face with atrophy of the muscles and a demarcation line between normal and abnormal skin. The right eye was mildly depressed in the socket, with asymmetry of the pupils and less prominent eyebrows. There was malar hypoplasia and atrophy of facial skin all confirmed by MRI. In addition, there was loss of subcutaneous fat with prominent bony ridges on the affected side and increased pigmentation on the ipsilateral upper forehead. There was atrophy of the right ala region of the nose (). A bald area can be seen on the scalp (). Other aspects of her clinical examination were normal. Based on this constellation of clinical features she was diagnosed with PRS/ECDS.\nInvestigations showed that hemogram, international normalized ratio, prothrombin time, partial thromboplastin time, renal, thyroid and liver function and urine analysis were normal. The antinuclear antibodies showed a fine speckled and homogenous pattern at 1/10 dilution: double stranded DNA antibody, rheumatoid factor, C3, C4, p and c antinuclear cytoplasmic antibodies, autoantibodies to extractable nuclear antigens, homocysteine and erythrocyte sedimentation rate were within normal limits. Cerebrospinal fluid analysis was normal with no oligoclonal bands. Abdominal ultrasound with doppler of renal arteries was normal. Echocardiogram was normal with an ejection fraction of 65%. An interictal electroencephalogram was normal.\nCT brain imaging showed areas of right parietal lobe hyperdensities consistent with calcification and mild atrophy of the right cerebral hemisphere as evidenced by prominence of the cerebral fissures and Sylvian fissure ().\nT1 FLAIR MRI axial views showed periventricular hyperintensities consistent with leukoariaosis more prominent in the right cerebral hemisphere with right frontal, temporal and parietal cortical atrophy (). Sagittal T2 FLAIR MRI view showed similar hyperintensities involving the corpus callosum (). Magnetic resonance angiogram of the brain and MRI spine were normal. Skin biopsy of an area of alopecia showed sclerosis of the dermis with normal adnexal structures and minimal inflammatory infiltrates of the mid-dermis.
A previously healthy 42 years old male presented to our institute with history of gradually progressive and painless swelling over left calf since two months. He was a non-smoker, laborer by occupation. The patient noticed a firm swelling in calf region of left leg 5 years ago. No history of trauma or infection prior to the appearance of the mass was reported. No family history of any such swelling in the past. Patient was operated for swelling over calf region 20 years back but no records were available. There was a history of gradual increase in deformity of left foot since 3 years and the patient had started walking on toes on left side with no dorsiflexion at ankle joint.\nOn examination, there was a single, non-tender, hyperpigmented scar of size 8×3 cm over mid-calf region fixed to underlying structures. A large, well defined non-tender, firm, swelling was palpable in posterior aspect of left leg measuring about 28×8 cm extending from tendoachilles region up to 5 cm distal to popliteal fossa and medially and laterally up to border of tibia and fibula respectively. The overlying skin was normal with no discoloration and local raise of temperature. Movement of knee joint was normal. There was fixed equinus deformity of left foot (). No inflammatory signs, skin changes or adenopathies were present. No bruits were heard on auscultation. Neurovascular examination of left leg and foot was normal. Laboratory findings were within normal limits. Radiological examination revealed large soft tissue mass with linear and streak-like ossification around the left tibia. MR Angiography () showed arteriovenous malformation in left calf with multiple feeding arteries arising from popliteal, peroneal and anterior tibial artery and large draining veins draining deep into venous system of leg. The tibia and fibula marrow showed normal signal intensity.\nBecause of patient symptoms and with clinical diagnosis of a vascular malformation, a wide surgical excision of the lesion was done. Through a 25 cm longitudinal incision across the calf, posterior compartment muscles were exposed. The mass was found completely involving superficial group of posterior compartment muscles sparing the deep compartment with no attachment to periosteum or bone (). Peroneal artery and vein were found to be embedded in the lesion and thus sacrificed. Plane of dissection was between superficial and deep muscles.\nThe lesion was completely removed along with overlying cutaneous scar with wide surgical margins leaving posterior tibial artery in continuity. Intraoperative, complete dorsiflexion of foot was achieved with intact vascularity of leg. The excised specimen was very hard like bone and had to be cut longitudinally with saw (). Grossly the resected specimen showed ossified tissue covered with skin and soft tissues including muscle, tendons and adipose tissue measuring 15×7×5 cm. The cut surface of the ossified area was grey white, gritty and congested (). Microscopically, it revealed features of a vascular malformation with numerous blood vessels of variable size and shape composed of arteries and veins which were dissecting soft tissues and interstitial planes of skeletal muscle.\nMany of the vessels were thin walled with anastomosing and a sinusoidal appearance. Some of them showed fresh and organized thrombi within this vascular background, extensive osseous metaplasia characterized by mature lamellar bone formation was seen. (). The diagnosis was consistent with arterio-venous malformation with extensive osseous metaplasia. At the time of recent follow up after one year from the operation, no local recurrence of the tumor was demonstrated clinically and radiologically. No restriction of motion of ankle joint was found. Patient is presently walking with a normal gait.
A 6-hour-old female neonate born from a 22-year-old Para 1 mother after term pregnancy. The newborn had protrusion of mass per vagina since birth; she also has a defect at the lower back with no discharge since the time of birth. Additionally the mother has noticed deformity on both legs and feet which barely move. The baby was active since the time of delivery and is sucking well and she passed meconium. The mother had two ANC visits and was vaccinated according to the national schedule and she reported the whole course of the pregnancy as uneventful. She did not have any known medical illness and has never taken any medication during the entire course of the index pregnancy except iron folate which was prescribed during the ANC visit.\nShe had spontaneous onset of labor and the membrane was ruptured spontaneously intrapartum. She gave birth to a 2330-gram female neonate after 5 hours of labor at a local health center. Baby had good APGAR score. Health care providers at the health center noticed defect at the back of the baby and referred her to Hawassa University Comprehensive Specialized Hospital (HUCSH) with the diagnosis of spinal bifida.\nDuring the initial evaluation at HUCSH the baby was active, vital signs were in the normal limits, and all neonatal reflexes were intact. There was 4x4 cm pink mass protruding through the introitus, cervical os is noted at the tip of the mass, the external genitalia appears normal, no discharge or bleeding from the mass, and the mass was reducible digitally and increases in size when the baby cries (see ). There was also 4x4 cm defect at the lumbosacral region the major portion of which is covered with skin while the lower edge is open, no discharge from the mass (see ). Additionally the newborn had bilateral club foot deformity (see ).\nOn investigation, the complete blood count was normal, creatinine level was 0.4mg/dl, and transfontanelle ultrasound scan shows mild dilatation of the lateral and third ventricles with an index of “mild hydrocephalus” and abdominal ultrasound was normal.\nAfter obtaining consent from the parents, under aseptic technique, the baby was catheterized, the vaginal mass was reduced digitally, and bandage was applied from the lower abdomen, both buttocks and legs were strapped in the bandage to the level of the mid-thigh leaving an opening at the anal orifice for passage of stool (see ). The bandage was removed after 72 hours at which time the mass was completely reduced and there was no recurrence of the prolapse afterwards (see ). Unfortunately one day after the removal of the bandage the baby started to shoot fever and had difficulty of sucking, on examination she had tachycardia and tachypnea and was febrile, she had depressed reflexes, the lower border of the meningocele got ruptured, her fontanels were bulged, and she was diagnosed with ruptured myelomeningocele and meningitis. The plan was to start her on antimeningitis drugs and repair the spinal cord defect after improvement but the parents insisted and went home against medical advice frustrated by her multiple anomalies. We communicated with the parents after her discharge and were informed that the baby died 7 days after she went home, but there was no recurrence of the prolapse throughout her stay.
An 18-year-old Caucasian woman with a history of NF-1 presented to the emergency department with pain in her left hip following minor trauma. She had tripped over her dog and landed on her left side. NF-1 had been diagnosed clinically in childhood. She had a histologically proven neurofibroma excised from her right forearm four years previously. She had a strong family history of NF-1, her mother and three second degree relatives exhibited clinical features.\nOn examination, we saw that her left leg was shortened, internally rotated, and adducted. There was decreased range of movement. She had diffuse swelling of her left lower limb with a distinct soft tissue mass above her right lateral malleolus. She had six cafβ-au-lait patches on her trunk and bilateral axillary freckling. A radiograph of the pelvis revealed a superior dislocation of her left hip with an abnormal appearing femoral neck (Figure ).\nHer hip was relocated under general anaesthetic and this was maintained with skin traction. A computed tomography (CT) of her pelvis displayed a smooth erosion of the lateral margin of her left ileum and femoral neck, markedly increased femoral neck offset, a concave abnormality superior to the left acetabulum and thinning of the left inferior pubic ramus. These changes, which appeared to be long standing, were accepted to be a result of a local neurofibroma causing bone erosion. Eight days post-operatively she experienced a sudden onset of left hip pain on attempting to move in bed. A radiograph of her hip revealed repeat dislocation. Relocation of her hip was performed under general anaesthetic and balanced skeletal traction was maintained by inserting a pin to her left proximal tibia. Skeletal traction was maintained for six weeks. She was mobilizing without aids at eight weeks and follow-up at three months revealed a normal hip examination.\nShe presented to the orthopaedic clinic six years later complaining of disfiguring hypertrophy of her left lower limb which had worsened markedly over the preceding two years. She was referred to plastic surgeons with a view toward performing a cosmetic debulking procedure. A pre-operative magnetic resonance imaging (MRI) scan of her lower limb demonstrated soft tissue swelling of her entire lower limb consistent with plexiform neurofibromatosis (Figure ). An MRI of her pelvis displayed a 6 × 4 cm enhancing mass at the superior aspect of the left acetabulum extending into the left hip joint (Figure ). There was smooth erosion of the neck of the femur but the head of the femur appeared to be in joint. An MRI of her lumbar spine was normal. A CT-guided biopsy of this hip lesion histologically confirmed it to be a neurofibroma. CT reconstructions demonstrated no further changes to the bone architecture of her left hip (Figure ). In view of her lack of symptoms and the degree of operative difficulty expected, the peri-articular neurofibroma was not excised. She underwent several debulking procedures of her lower limb with excellent cosmetic results. At 12 years follow-up she has experienced no further dislocations of her left hip and mobilizes without aids with a normal gait. A radiograph of her pelvis demonstrated her left hip to be in joint (Figure ).
A 46-year-old woman with morbid obesity, hypertension, hyperlipidemia, active smoking, and bipolar disorder was evaluated for paroxysmal atrial fibrillation as an outpatient. A transthoracic echocardiogram showed a right atrial mass close to the RA-IVC junction. A transesophageal echocardiogram confirmed the presence of a pedunculated right atrial mobile bilobular mass measuring 2.1 × 1.8 cm, not arising from the interatrial septum with extension into the inferior vena cava (Figures –). A secundum atrial septal defect was also identified. The patient was referred to cardiac surgery for resection of a presumed right atrial myxoma given the possibility of embolization. The intraoperative TEE suggested the presence of one bilobulated mass arising low in the right atrium.\nUsing aortic and bicaval cannulation, a right atriotomy was performed and a discrete 2 × 3 cm mass was removed from the IVC-RA junction near the Eustachian valve. The right atrium was closed and the patient weaned from bypass. The specimen did not appear to be a myxoma, so it was sent for frozen section and interpreted as being ectopic liver tissue. In the interim, a repeat TEE showed an additional discrete 2 × 2 cm mass in the IVC near the hepatic veins. Femoral venous cannulation was then performed to allow for visualization of the IVC below the pericardial reflection. Through the same right atriotomy and using additional suction directly in the IVC, the IVC mass was visualized and resected. Both specimens were interpreted as showing partially encapsulated liver parenchyma with mild steatosis, fibrosis, ductal proliferation, and periductal chronic inflammation, consistent with ectopic hepatic tissue ().\nDespite a brief period of cardiopulmonary bypass, the patient had persistent hypoxemic respiratory failure ascribed to her morbid obesity and underlying lung disease. She also developed extensive left lower extremity deep venous thrombosis from a presumed heparin-induced thrombocytopenia for which she was treated with argatroban and placement of an IVC filter. Because of chronic respiratory failure, she underwent tracheostomy and feeding tube placement, and she was placed on warfarin for intermittent atrial fibrillation due to her CHA2DS2-VASc score of 4. She gradually improved and was transferred to rehab where her tracheostomy tube and PEG tube were removed. A few months later, she looked quite well during an office visit.
A 27-year-old male, married, graduate, presented with a history suggestive of OCD since the age of 19 years. The disorder was characterized by having obsessive images of known people, leading to significant anxiety and distress. To get rid of his anxiety and distress, the patient would repeat the acts at hand, such as crossing the doors and washing hands, multiple times till these images would subside or till he was able to replace these images by desirable images. These symptoms led to marked socio-occupational dysfunction to the level that he was not able to work and suffered losses in his business. There was no history suggestive of any comorbid physical or psychiatric illness including tics. His treatment history revealed that he had received adequate trials of multiple selective serotonin reuptake inhibitors (SSRIs) and currently was on capsule fluoxetine 80 mg/day with minimal improvement. Evaluation also revealed that in the past, ERP was tried by using imaginal exposure, but this was unsuccessful, as the patient would avoid imagining the anxiety-provoking images.\nIn view of the treatment resistance, he was considered for inpatient management. Initially, behavior analysis was done and behavior therapy in the form of ERP was planned.\nThe behavior analysis also involved construction of the hierarchies for exposure. Two separate hierarchies with ascending order of subjective units of distress were made. First hierarchy was created with the pictures of the persons which caused the distress, while the second hierarchy was created according to the tasks that the patient would repeat on experiencing obsessive images.\nBefore starting behavior therapy, the patient and his spouse were psychoeducated about the illness and treatment. This involved explaining them the causes, signs and symptoms, treatment options for OCD, and principles of ERP. In addition, the patient was taught Jacobson's Progressive Muscle Relaxation (JPMR) technique.\nFor the ERP, pictures of the persons whose images caused distress to the patient were collected. These pictures were incorporated in videos containing pictures of other people (known and unknown to the patient) that presented the anxiety-provoking images randomly. With each image, the name of the person (i.e., image presented) was recorded in patients' own voice and the same was also presented as part of the video used for exposure. The videos were of 15 min duration, which included images and audio recordings of names, played in a loop. Exposure was started from the least anxiety-provoking images clubbed with the hierarchy of tasks. The patient was made to watch the video by using the virtual reality headset and simultaneously made to do the task at hand as per the hierarchy. His parameters (i.e., vitals and subjective unit of distress) were noted every 5 min till the anxiety came down to zero. Each session lasted for about 45 min to 1 h. After all the task with one set of images was completed, we moved on to the next task in the hierarchy. Over a period of 2 months, 60 sessions of ERP were carried out and the whole hierarchy was completed. Gradually, he went through the whole sequence of images and videos, which were clubbed with response prevention. The experience of the patient was very satisfying as this kind of exposure was more close to his experience of obsessive images, and according to him, this helped in getting better with behavior therapy.\nWith each increasing step, for generalization, exposure to all the previous images along with addition of new images was continued. By the end of the therapy, his Yale Brown Obsessive Compulsive Scale severity score came down to 5 from baseline score of 28. During the whole therapy, the patient was allowed to practice JPMR, although this was not clubbed with ERP sessions. In addition, supportive psychotherapy and booster psychoeducation sessions were continued. In addition to the virtual reality exposure, he was also made to write the names of the people, whose names would provoke anxiety.\nDuring the follow-up, booster sessions were carried out. The patient is currently maintaining 80% improvement in symptoms with no dysfunction in work.
A 54-year-old woman visited our department due to bilateral hearing impairment and intermittent otorrhea without dizziness of protracted duration. She did not have tinnitus or otalgia, but complained of recent symptom aggravation. Furthermore, she had no known history of medical or surgical problems, except that she was a chronic hepatitis B carrier.\nDuring physical and radiological examinations, a yellowish purulent discharge was noted in the right ear canal and attic destruction was identified. The left ear drum was thinned inferiorly and in a retracted state (), and the Weber test showed left deviation. Pure tone audiometry showed air conduction threshold of 72 dB HL and bone conduction threshold of 28 dB HL for the right ear and air conduction threshold of 50 dB HL and bone conduction threshold of 20 dB HL for the left. Auditory brainstem response testing showed peak waves at 70 dBnHL on the right and at 50 dBnHL on the left. A temporal bone computed tomographic (CT) scan showed sclerosis of mastoid bone and soft tissue densities in the middle ear, attic area, and mastoid cavity on the right side (), but ossicles and other inner ear structures, and the external auditory canal were relatively healthy. Other routine laboratory tests were within normal ranges. However, no evaluation of cerebrovascular structures was performed.\nTwo days after admission, right side canal wall up mastoidectomy with type I tympanoplasty was performed under general anesthesia. The dura and sigmoid sinus were not exposed during surgery. At about 2 hours after surgery, the patient complained of a headache and a rigid sensation in upper and lower extremities. At this time, her blood pressure was elevated at 170/100. Mentally, she was mildly drowsy and oxygen saturation had fallen to 80%. Accordingly, she was placed on mask ventilation, and our department of internal medicine was consulted regarding management. Endo-tracheal intubation was decided upon but not instituted because her condition recovered to near normal within an hour. Department of neurosurgery was then asked to undertake a proper evaluation. However, brain CT returned non-specific findings. Subsequently, the patient was followed a normal recovery course and pattern. Nevertheless, we recommended further evaluation, particularly brain magnetic resonance image, but the patient and her family rejected the idea. Accordingly, regular discharge was ordered 1 week after surgery.\nTwo weeks after discharge, the patient visited our emergency room with a severe headache and a mental status of stupor. Emergent brain CT and angiography was performed and a subarachnoid hemorrhage (SAH) from a ruptured anterior intercommunicating artery was visualized (). Emergent trephination was performed, and subsequently, she was admitted to our neurologic intensive care unit. Nevertheless, intracranial bleeding persisted at follow-up brain CT 2 days later (). The patient expired 1 week after this second admission without recovering consciousness.
A 37 year old man arrived at the outpatient facilities of our clinic complaining of a remittent pain at the right lateral abdomen and a palpable mass that was constantly growing up during the last twelve months. His medical history included an uneventful appendicectomy in his childhood, 27 years ago and spontaneous pneumothorax 15 years ago. By that time, he was under medication with antacids because of gastritis. He did not mention any evacuation or urination symptoms.\nPhysical examination revealed a firm, sizeable and hard mass at the right abdomen (Figure ), extending from the inferior ribs to the right iliac crest. Laboratory findings were within normal limits. The ultrasound (US) of the abdomen demonstrated a compact mass of uneven shape at the right abdomen, extended from the liver to the minor pelvis. Abdominal computed tomography (CT) scan, demonstrated a cystic textured lesion, extended from the lower pole of the right kidney to the right iliac fossa, with a cefalocaudal diameter of 11 cm (Figure ). There were small diaphragms within the mass, enforced by the intravenous administration of a contrast essence. The tumor was expelling not only the homolateral ureter but also the small and the large intestine to the left, however, without any findings of obstruction. The magnetic resonance imaging (MRI) of the abdomen demonstrated a cystic tumor with diaphragms (Figure , Figure ), extending along the right abdomen from the inferior pole of the right kidney to the right iliac crest. The tumor measured a maximum cefalocaudal diameter of 22 × 10 cm, causing a slight pressure of the inferior vena cava. The administration of paramagnetic essence revealed an uneven peripheral enhancement of signaling. Three focal lesions of magnetic abnormal signaling were brought out at the liver segments II, VI, VII, and VIII, suggesting secondary metastasis or hemangiomas. Colonoscopy and upper GI endoscopy was carried out in order to exclude involvement of the gastrointestinal tract. Gastritis was the only abnormal finding.\nThe patient underwent laparotomy. A retroperitoneal cystic mass of gelatinous-like content, arising from the paravertebral space of the L1-L2-L3 vertebra, was confirmed. An "en block" resection of the tumor was performed. The small and the large intestine as well as the right ureter did not present signs of primary or metastatic involvement.\nThe first postoperative hours, the patient complained of sub-hyperesthesia at the inner surface of the thigh that was extended to the middle of the calf. In addition, he asserted a reduction of muscle power at his crur (thigh flexion debility). Within the next 24 hours, he underwent an electromyogram which demonstrated a possible neurogenic damage, due to lesions of the L2-L3 level. Methylprednisolone (250 mg) was administrated as a single boost dose, followed by dexametasone (8 mg three times a day). The patient was discharged the fourth postoperative day without any other postoperative complication.\nThe tumor weighed 957 g and measured 22 × 14 × 4,5 cm. The histological examination described internal cysts within the mass, measuring from 1 to 4 cm in diameter. The thickness of the cystic wall was variable (from 0,1 to 0,5 cm). Microscopically the inner surface of the cystic wall revealed filiform and branching papillae lined by mucin-containing atypical epithelial cells (Figure ). The lining cells were stratified, generally to two or three layers, and the nuclear atypia was mild to moderate. The collagen fibers of the wall were disintegrated by pools of mucous, epithelial cells and calcifications. Lymphocytic infiltration was also observed. The PAS-D and Alcian Blue stain was positive as well as the immunihistochemical stain for keratine 8/18, keratine 20, pankeratine, CEA and Ki-67 (Figure , Figure ).\nTwo months later the patient had a new abdominal CT-scan which demonstrated absence of residual disease. The electromyogram was compatible with a chronic neurogenic damage of the quadriceps muscle. The next two abdominal CT-scans, performed 6 and 12 months later, showed no recurrence. A significant clinical improvement was observed in patient's neurological status, followed by a considerable increase of muscle power.
A 28 year-old male was found to have a slightly increased serum creatinine (1.7 mg/dl) during the admission to ER because of abdominal pain. Since both his past medical history and an abdominal ultrasound performed on admission were unremarkable he was discharged with the indication to see a Nephrologist. At the Nephrology outpatient clinic, because of the reduced eGFR and of the presence of proteinuria (300 mg/24 h), an hospitalization for further investigations was planned.\nWhile waiting for the admission, a couple of weeks later, the patient was admitted to the emergency room in another hospital for abdominal pain and dyspnea. On admittance the ECG tracing performed showed sinus rhythm with diffuse repolarization abnormalities. Further exams revealed an increase of troponin (54 ng/ml) and transaminases levels (GOT 50 U/L; GPT 125 U/L) and a cardiac ultrasound showed a severe left ventricular dysfunction with right heart failure. Patient was thus admitted to the coronary intensive care unit. During his hospital stay the possibility of acute myocarditis was excluded because of the absence of a recent viral syndrome and the negativity of IgM antibodies against the viruses most commonly affecting the cardiovascular system and because of the low inflammatory indexes. An abdominal ultrasound demonstrated abdominal and pleural effusions, regular size and morphology of spleen and liver, no pancreatic changes; both kidneys had normal size but showed irregular margins and a reduced thickness of the cortex, no hydronephrosis nor stones were observed. A magnetic resonance demonstrated an increased volume of the cardiac chambers with a severe reduction of the function of both ventricles (EF20%), however no signs of reduced myocardial perfusion, valvular defects or other tissue changes such as inflammation, fibrosis or edema could be demonstrated. A chest CT scan confirmed the presence of pleural effusion and showed parenchymal atelectasias mainly involving the lower lobes of the lungs. The patient was discharged after 11 days on diuretics (furosemide 100 mg/daily), B-blocker (carvedilol 37.5 mg/daily), acetyl salicylic acid (Aspirin 100 mg/day) and an Ace inhibitor (Ramipril 5 mg/day) with a diagnosis of dilated cardiomyopathy and severe left ventricular dysfunction complicated with cardiogenic shock and anasarca. Few days later an EKG ergometric test revealed no areas of inducible ischemia. A further admission to the Cardiology Unit to define the effect of treatment was performed a week later. Cardiac ultrasound confirmed the left ventricular dysfunction (EF 23%), a coronary angiography was negative and a right chamber catheterism and endomyocardic biopsy demonstrated no specific lesion nor inflammatory infiltrate. The patient was then admitted to our Nephrology Unit, as planned, to evaluate the cause of the chronic kidney disease. On admission the patient appeared healthy and well oriented, his blood pressure was 110/60 mmHg, heart rate 56 bpm, physical examination was unremarkable except for a 1/6 intensity systolic heart murmur better audible on mitral valve area. ECG tracing showed sinus bradycardia. Laboratory studies revealed an increased serum creatinine (1.87 mg/dl), eGFR 48 ml/min/1.73 m2, hypercalcemia (serum calcium 11.5 mg/dl), hemoglobin 15.1 g/dl, sodium 135 mEq/L, potassium 4.37 mEq/L, phosphate 3.1 mg/dl, serum albumin 4.07 g/dl. The patient denied use of vitamin D analogs thus in consideration of the unexplained hypercalcemia other exams were performed. Thyroid hormones, calcitonin, vitamin D, serum ACE, phosphate and calcium urinary excretion rate were all within the normal range. PTH was lower than 3 pg/ml. To rule out multiple myeloma serum protein electrophoresis, serum and urine immunofixation, and spine and skull x-ray were performed and did not show any pathological changes.\nRenal biopsy was finally performed without complications and the patient was discharged after 24 h. While waiting for the renal biopsy pathology report, medications were not modified, but for an increase of the dose of furosemide because of the hypercalcemia. The renal biopsy contained eight glomeruli none of which was globally or partially sclerotic and no pathological changes were observed at glomerular level, vessels were also unremarkable. Tubulointerstitium was the only affected compartment with a moderate to severe inflammatory infiltrate, some calcium crystals in the tubular lumen and two granulomas with multinucleated giant cells (Figs. and ). No necrosis could be observed within the granulomas. A diagnosis of granulomatous interstitial nephritis due to sarcoidosis was made.\nWhen the patient was called to discuss his kidney biopsy, his serum creatinine had further increased (3.5 mg /dl), eGFR 22 ml/min/1.73 m2 and so had his serum calcium (13.3 mg/dl), serum albumin 4.06 g/dl. For this reason, he was hospitalized again and treated with pulse methylprednisolone (250 mg/day for three days) and then with maintenance dose prednisone (50 mg/day). Serum creatinine decreased in a few days and so did his serum calcium. Since the episode of acute kidney injury the patient has been followed at our unit and at the unit of Cardiology. Renal function returned to normal (serum creatinine 1.1 mg/dl, eGFR 91 ml/min/1.73 m2) and heart function markedly improved over the following two years (EF 49% vs 37% vs 32% vs 23%). The diuretic and cardiologic therapy was significantly reduced to only 25 mg of furosemide and a small dose of beta blocker. The steroid was gradually tapered to 5 mg/day of prednisone for one year and then stopped.
A 58-year-old woman with an unremarkable medical history visited the emergency department (ED) for worsening dizziness and dyspnea, noted one day before. The patient was alert and had normal peripheral oxygen saturation (SpO2) when the paramedics arrived. However, her mental state and oxygen saturation level declined during transport to the hospital. She had an SpO2 of 83% despite receiving 100% oxygen supplementation through a face mask with a reserve bag. The blood pressure was not measured due to a weak pulse. The patient was drowsy upon arrival at the ED. Cardiac arrest with pulseless electrical activity occurred after five minutes, and spontaneous circulation was restored after two minutes of cardiopulmonary resuscitation. A computed tomography (CT) of the brain, chest, and abdomen–pelvis (AP) was taken two hours after arrival. The AP-CT revealed an 8 × 11 cm saccular aneurysm involving the left and right renal veins at the infrahepatic level of the IVC (). The chest CT revealed massive pulmonary embolisms involving both distal main pulmonary arteries (a). A hyperdense middle cerebral artery (MCA) sign indicating thrombosis of the M1 MCA segment and widely decreased gray matter densities in the left hemisphere and right frontal lobe were detected on the brain CT (b) []. A tissue plasminogen activator was administered two and a half hours after arrival, and the patient was admitted to the intensive care unit. The patient developed a seizure; therefore, a follow-up brain CT was performed after six hours. This showed a broad infarcted area involving most of the cerebrum, except for the thalamus and right occipital lobe. An electroencephalogram taken two days after admission suggested the possibility of brain death, and a bedside transthoracic echocardiography performed on the fourth day of hospitalization showed an enlarged RV with reduced RV systolic function. The patient’s family decided against a further evaluation or advanced treatment; therefore, the patient was conservatively managed at this point. Normal blood pressure was not achieved despite administering high-dose inotropic agents during the seventh day of hospitalization, and the patient died on the eleventh day of hospitalization.
At the beginning of 2000, a 52-year-old Caucasian man who worked as a dental surgeon and had never smoked, was referred to the our Occupational Health Unit, owing to shortness of breath during exercise, cough and retrosternal pain, which had become progressively evident in the months prior to admission. His medical history was unremarkable. Physical examination, pulmonary function tests and blood tests were within normal ranges. Chest X-ray showed bilateral hilar enlargement and suggested hilar lymphadenopathy secondary to sarcoidosis as a possible diagnosis. This possibility was supported by chest computed tomography (CT) scan, which revealed diffuse hilar and mediastinal lymphadenopathy with maximal lymph node diameter of 3 cm. Total body scintigraphy with gallium 67 confirmed the hilar and mediastinal lymphadenopathy. Bronchoalveolar lavage (BAL) showed an increase in total cell count and an elevated CD4/CD8 ratio compatible with sarcoidosis, and absence of neoplastic cells or infectious agents. Finally, cytologic examination of the aspirated bronchial material evidenced a non-caseating granuloma and first stage sarcoidosis was diagnosed. After lung examination, it was decided not to start any treatment and to evaluate the progression of the disease. Therefore, our patient was referred for follow-up.\nGiven the epidemiologic evidence on the role of occupational exposure in the etiology of sarcoidosis, a sample of the powder used by our patient in his work as a dental surgeon for at least 20 years in dental hygiene procedures was analyzed. Analysis was performed by electronic microscopy in the Laboratory of Biomaterial of the Department of Neurosciences, University of Modena and Reggio Emilia and identified the presence of silicates among components.\nDuring the following three years, the clinical picture and chest CTs remained unchanged. Blood tests showed an increase of serum angiotensin converting enzyme (ACE) and GPT, whereas calcium remained within normal values. Hepatic echography was also normal. In 2003, our patient's dyspnea was seriously aggravated with episodes also occurring at rest; the chest CT performed in March 2003 revealed a bilateral increase in the number and volume of the affected mediastinal and hilar lymph nodes, whereas the remainder of the pulmonary parenchyma was normal. Consequently, treatment was initiated with cycles of cortisone, which produced visible improvement in the clinical picture, reduction of the lymphadenopathy on CT after seven months, and decline in serum ACE to within normal range. In 2006, while our patient's dyspnea remained under control with cortisone, CT showed progression of the disease with enlargement of the hilar and mediastinal lymphadenopathy and areas of interstitial thickening. Additional information about the possible origin of the disease was provided by SEM with X-ray microanalysis of biopsy specimens, which showed that the material found inside the sarcoidotic granulomas was identical to that found in the powder used by the dental surgeon for several years. Currently, our patient is working without using any dust for dental cleaning, his clinical picture is stable with daily cortisone treatment, and he is under periodic control.
A 55 year-old man visited our emergency department because of increasing frequency of chest pain. He had undergone off-pump coronary artery bypass grafting (CABG) 10 years ago because of unstable angina associated with three vessel coronary artery disease. At the initial operation, the in situ right internal thoracic artery (ITA), in situ left ITA and in situ right gastroepiploic artery (RGEA) grafts were used to revascularize the left anterior descending coronary artery, two obtuse marginal coronary branches, and posterior descending coronary artery, respectively. An excess segment of the distal right ITA was connected to the side of left ITA as a Y-composite graft and anastomosed to the first diagonal coronary artery. Coronary angiography and myocardial single photon emission computed tomography (SPECT) were performed at 5 years after surgery as a follow-up study. The 5-year angiography showed all patent grafts and the myocardial SPECT demonstrated no perfusion decrease. Exertional chest pain recurred at 7 years after surgery, and a repeated coronary angiography showed patent previous grafts including faint visualization of the in situ RGEA graft associated with significant stenosis at the os of the celiac axis. The computed tomographic angiogram also demonstrated a 90% stenosis at the celiac os, which had been without stenosis on abdominal angiography taken before the surgery (). The myocardial SPECT, however, demonstrated no perfusion decrease. Redo off-pump CABG was performed 10 years after the initial surgery because of an increasing frequency of angina and an aggravated finding of the follow-up myocardial SPECT, which was a newly developed reversible perfusion decrease in the inferior wall ().\nAt reoperation, the great saphenous vein was harvested from the lower leg and interposed between the middle part of in situ right ITA and distal part of in situ RGEA grafts used previously, to supply blood flow from the right ITA graft to the posterior descending coronary artery. The patient was discharged without any complication on the 9th postoperative day. One year after redo surgery, the patient had no symptoms of angina and coronary angiogram was performed and revealed patent grafts, including an interposed saphenous vein graft (). The myocardial SPECT test was also performed and demonstrated that there was no perfusion decrease including the inferior wall ().
The patient involved provided written consent for reporting of this case. A 63-year-old woman with medical history of super morbid obesity (BMI 54) and atrial fibrillation for which she was anticoagulated with apixaban presented for an open reduction internal fixation (ORIF) of an ankle fracture. Significant medical history included diabetes mellitus type 2, obstructive sleep apnea, chronic obstructive pulmonary disease, and diastolic heart failure. The patient's last dose of apixaban was 48 hours prior to surgery. Other than moderate anemia (hemoglobin 8.8 g/dL), all laboratory studies, including a coagulation profile, were normal. Prior to surgery, the patient was offered a sciatic nerve catheter and an adductor canal block as part of a multimodal postoperative analgesia strategy. Because of her many, serious medical conditions, we concluded that a peripheral nerve block offered the best opportunity to provide satisfactory postoperative analgesia. Specifically, we were concerned that the postoperative pain management primarily with opioid medications would pose increased cardiopulmonary risk to the patient. We were careful to explain the risks associated with peripheral nerve blocks, including the risk of bleeding and hematoma formation, and verbal consent was obtained. The surgery was performed under general anesthesia and her intraoperative course was uncomplicated. Upon arrival to the recovery room, our acute pain service was contacted to evaluate her for peripheral nerve blockade. We positioned the patient in the lateral decubitus position and placed a sciatic nerve catheter. Though technically challenging due to body habitus, this sciatic nerve block was performed successfully and without any complication. The patient was then positioned supine for the adductor canal block. The leg was externally rotated and the knee slightly flexed for optimal positioning. A high-frequency linear array ultrasound transducer was applied to the mid-thigh in short-axis and the adductor canal was identified. Imaging was again challenging given the patient's habitus, but with firm compression of the ultrasound transducer, the important anatomical structures were clearly identified. The superficial femoral artery (SFA) was visualized
An 18-year-old male “Wushu” martial arts athlete presented to our outpatient clinic with a palpable right inguinal mass and claudication for 6 months. His past medical history was unremarkable. On physical examination, a nontender, firm mass measuring 2 cm was palpable in the right inguinal area and the right ankle pulse was not palpable. The ankle-brachial index (ABI) was measured as 0.57 on the right side and 1.13 on the left side. Duplex ultrasonography revealed a cystic lesion that was compressing the common femoral artery (). Computed tomography (CT) revealed a cystic lesion located superomedially to the right common femoral artery causing severe stenosis (). Magnetic resonance imaging also showed the cystic lesion and its communication with the right hip joint ().\nPreoperatively, a GC of the hip joint was suspected and an elective operation was performed. A longitudinal incision was made to the right groin, and the common femoral artery and surrounding tissue were dissected carefully. However, dissection of the arterial wall to the lesion was impossible, and we suspected ACD of the femoral artery at this point. We performed a stab incision of the cyst, and gelatinous material was extracted from the cyst (). The entire length of the cyst (4 cm) was inspected through a longitudinal incision, the cyst was excised, and the connection to the hip joint was ligated. After excision of the cyst, we observed the medial layer of the femoral artery without any overlying adventitia, and we decided to insert an interposition graft with a prosthetic graft because of the friability of the remaining arterial wall and a small diameter of the saphenous vein. Histological examination of the surgical specimen demonstrated that the cystic space was surrounded by dense collagenous fibrous tissue without synovial cell lining (). Postoperatively, the patient recovered without complications and was discharged 6 days after the operation. His ABI returned to normal (0.99 on the right side and 1.12 on the left side) and the claudication disappeared.
A 24-year-old lady presented with mild pain in the right lumbar region. The physical examination was unremarkable. Standard laboratory test results, including urinalysis and urine culture, were within normal range. Her past medical history included a rhabdomyosarcoma of the right buttock surgically treated at the age of six months and a germiline pathogenic p53 mutation (Li-Fraumeni syndrome). She did not take any regular medication and she was in good general health. An abdominal ultrasound scan revealed two uncertain-in-nature lesions, one within the right part of liver and one within the upper pole of the right kidney.\nA subsequent MRI scan of liver and kidneys confirmed the presence of the two lesions that were shown at ultrasound scan. The lesion within segment 7 of the liver was 1,8 × 1,3 cm in diameter. It was solid and showed restricted diffusion. It was enhanced early and fairly intensely following contrast administration and also exhibited early washout (). The lesion within the upper pole of the right kidney showed peripheral enhancement following contrast administration and demonstrated impeded diffusion at its peripheral aspect ().\nThe MRI features of the two lesions were not pathognomonic for a specific pathologic entity. Following discussion within the liver and urology multidisciplinary team meetings, a recommendation for simultaneous right partial nephrectomy and partial hepatectomy was made (due to the patient's Li-Fraumeni syndrome and the associated high risk of malignancies development).\nThe patient underwent an uncomplicated open simultaneous right partial nephrectomy and resection of segment 7 of the liver and she was discharged 5 days later.\nThe morphological and immunohistochemical features of both lesions were of epithelioid angiomyolipoma (PEComa).\nHistopathological examination revealed that both lesions were confined to the liver and kidney, respectively, with no evidence of invasion into surrounding tissues. The border between the tumours and the surrounding liver and renal parenchyma, respectively, was well defined. The size of the kidney and liver lesions according to histopathological examination was 30 mm and 10 mm, respectively. There was no lymph node or lymphovascular invasion and both lesions were completely excised.\nBoth lesions were composed of an admixture of spindle and epithelioid cells, with a predominance of spindle shaped tumour cells (>70% of the tumour cells). Within both lesions there were scattered mature adipocytes and also scattered pleomorphic cells with anaplastic and multinucleated nuclei. Definite tumour necrosis could not be identified within either of the lesions.\nMitotic figures were easily seen only within kidney lesion, amounting to 10 per 10 hpfs with scattered atypical forms being present, while within liver lesion mitotic figures were not identified.\nImmunohistochemistry of both lesions shows the tumour cells to strongly express melan A, HMB45, smooth muscle actin (SMA), and caldesmon, with focal S100 expression. There was no expression of cytokeratin-AE1/AE3, cytokeratin-CAM5.2, CD10, CD117, or DOG1. Additionally there was no expression of EMA, PAX8, RCCAg, myogenin, and MyoD1 within the kidney lesion, and there was no expression of CK5/6, Hepar1, AFP, and CD34 within the liver lesion. The proliferation marker Ki67 was expressed in approximately 10 to 20% of the tumour cells within both lesions. The only difference between the two lesions regarding immunohistochemistry was the absence of desmin expression within the liver lesion while there was focal desmin expression within the kidney lesion.\nThe patient did not receive any adjuvant systemic treatment and, at the last follow-up, 1 year from surgery, she remains disease-free.
The patient was a 34-year-old woman, G4 L2 Ab1 who had married her cousin 7 years ago. She had a history of two normal vaginal deliveries and one abortion in the 1st trimester. The first pregnancy in the age of 28 had terminated with a normal vaginal delivery (NVD) resulting in a term baby girl who weighed 3150 gr. The second pregnancy had occurred two years later; curettage was done at week 6 due to spontaneous abortion. Her 3rd pregnancy was in the age 32 resulting in a healthy term baby girl weighing 3400gr with NVD. Due to her unwillingness for becoming pregnant she had withdrawal contraception, whereas because of the non-occurrence of menstruation during breastfeeding and 6 months after her last pregnancy, a pregnancy test was requested. Due to the positive pregnancy result, ultrasound study was done which revealed a 10-week spontaneous gestation with 4 gestational sacs and 4 fetuses. There was no case of multiple pregnancies in her or her husband's family.\nShe received prenatal care during her pregnancy but there was no need for prophylactic cerclage. At week 24 of gestation she was hospitalized due to premature contractions. The contractions were controlled with the prescription of pethidine and hydration and she was discharged 3 days later. She was once again admitted at 28 weeks of gestation due to similar contractions; this time she was treated with indomethacin and pethidine and discharged 3 days after the contractions suppression. She also received two doses of betamethasone during hospitalization.\nShe was admitted a week later due to labour contractions. In vaginal examination 2 finger dilatation with no effacement was detected. Serum test results were reported all in the normal range and the vital signs during hospitalization were normal. At this stage she was treated with tocolytics (adalat). The fetuses' health was monitored by Doppler ultrasound imaging, biophysical profile and fetal non stress test (NST). After the labour contractions' suppression and due to the presence of sporadic contractions she was monitored while being hospitalized up to the time of delivery.\nAt 32 weeks and 4 days of gestation, due to the resumption of labour contractions and dilatation progression, after receiving the rescue dose of betamethasone, cesarean section and tubectomy (upon the request of the patient and her husband) was performed. The outcome of cesarean section was 4 fetuses, 3 girls and a boy, quadriamniotic and quadrichorionic. Quadruplet A weighed 1820 gram with an Apgar score of 9 to 10; quadruplet B weighed 1810 gram with an Apgar score of 6-7. Quadruplets C and D weighed 2100 and 1980 gram with an Apgar score of 7-8 and 9-10, respectively. Among the 4 neonates, only quadruplet B was transferred to the NICU; she was discharged after 2 days in good health. and show the quadruplets after birth.\nBecause of atonic uterus during the cesarean section, after the administration of the appropriate dosage of oxytocin and methylergonovine and 800µgr of rectal misoprostol, the uterine arteries were blocked and the B-Lynch suture was done. No blood transfusion was required for the mother and her hemoglobin (Hb) level 6 hours after the operation was 9 g/dl; her pre-operational Hb level was 10g/dl. The mother was discharged 3 days after delivery with no complications.\nFor close follow up, the mother and her newborns were visited two weeks after delivery; they were all healthy and had no problem. The infants were visited once again 6 months later revealing normal physical and mental development in all four. shows the babies at 6 months of age.\nThis project has been approved by Ethical Committee and Vice Chancellor for Research of Mashhad University of Medical Sciences (97/429008).
A 69-year-old woman presented to our institution with right abdominal pain. There was nothing special to mention in her family history. Her past medical history included horseshoe kidney, gastric ulcer, and asthma. Physical examination revealed a slight tenderness of the right quadrant abdomen. All laboratory parameters including the tumor markers carcinoembryonic antigen and carbohydrate antigen 19-9 were within normal limits. Abdominal ultrasonography showed a regularly shaped uniform tumor of about 50 mm in diameter that was located in the right retroperitoneum ventral to the right part of the horseshoe kidney. The tumor was hypervascularized (). Computed tomography (CT) showed a tumor of 80 mm in diameter ventral to the right part of the horseshoe kidney and the dorsal side of the descending part of the duodenum. On contrast-enhanced CT, the tumor showed late-phase enhancement. There were no findings of invasion into any organs and right ovarian vein ran through the tumor (). No metastases to organs or swollen lymph nodes were found. Magnetic resonance imaging (MRI) showed a tumor that was isointense with respect to muscle on T1-weighted images and of high-signal intensity on T2-weighted images (). No fatty components were detected in the tumor. Endoscopic ultrasonography showed a regularly shaped and hypoechoic tumor with no connection to the right part of the horseshoe kidney or duodenum (). Our working diagnosis was a retroperitoneal tumor that could be either a malignant lymphoma, leiomyoma or gastrointestinal stromal tumor.\nIntraoperatively, the tumor was located in the right retroperitoneal space and did neither adhere to nor invade other organs. The right ovarian vein ran cranially to caudally through the tumor. A central tumor thrombosis was detected in the ovarian vein (). We performed the resection of the tumor together with the right ovarian vein. The specimen showed a grayish-white solid tumor with the ovarian vein passing through its center (). Microscopically, fascicular hyperplasia of eosinophilic spindle cells with high-grade dysplasia and atypical mitotic figures were detected. Elastic fibers of the vessel wall were identified in the tumor. Immunostaining revealed that the tumor was positive for smooth muscle actin and desmin and negative for s-100 protein and c-kit. Based on these findings, our diagnosis was a LMS arising from the ovarian vein (). The immediate postoperative course was uneventful and the patient was discharged on day 11 postoperatively. Five months after the surgery, multiple lung metastases were detected on CT and chemotherapy was initiated (doxorubicin + olaratumab).
A 25-year-old female patient reported with a complaint of watery discharge from her right cheek region since last 1 year. Patient gave history of fever and malaise associated with swelling in the right cheek region, which increased in size gradually and did not decrease for the next 15 days. Patient applied balm and hot fomentation over the swelling and the swelling started draining with purulent discharge over the cheek region. Patient consulted a local physician and was prescribed some medications for 5 days subsiding the swelling and pus discharge. Since then, watery discharge started from the same region. According to the patient, the discharge increased during meals. Patient received consultation from various places for the same but did not get any relief. Patient also consulted a local dentist for the same complain and underwent surgical extraction of the lower right third molar, which was impacted. Even 10 days after-the extraction, patient had no relief and then she was referred to our institution.\nPatient gave no contributory medical history with a healthy familial background and no personal abusive habits. Extra-oral examination revealed a scar tissue on the right cheek on anterosuperior region of the angle of mandible, oval in shape and approximately 1 cm × 1.5 cm in size. The surrounding skin appeared to be normal. A small pinpoint opening () was seen just above the scar from which watery fluid discharged spontaneously which increased on sucking lemon. On palpation, all inspectory findings were confirmed. The scar was completely healed and attached to the underlying deeper structures. The discharged fluid was watery, non-sticky, and thin in consistency. Right submandibular lymph nodes were palpable, enlarged, freely movable and non-tender, measuring approximately 1 cm in size. Intraoral examination revealed healing extraction socket with 48 with no tenderness present on the same region. The parotid papilla appeared to be normal. Mild salivary flow was seen from right parotid papilla along with more flow from fistula on milking of right parotid gland and rest of the oral cavity showing normal wet ability with saliva ().\nUltrasonographic examination revealed normal echostructure of the right parotid gland with smooth margins, giving no evidence of any solid or cystic lesion. A small collection was seen in the anteroinferior margin of the right parotid, which measured 5 mm × 3 mm approximately. The contents were anechoic, and a small 1.5 mm sinus tract was seen from the collection to the skin.\nOn radiographic examination with insertion of gutta percha point in the fistula revealed in the parotid region (). Fistulography, as well as sialography, was performed using 70% urografin contrast media which revealed its relation with parotid duct. When the urografin solution was injected into the parotid duct intra-orally the solution came out from the fistulous tract extra-orally immediately without entering into the gland tissue. This was seen because of least resistance felt on this route. Hence, a false filling defect in radiograph was noted, and solution could not enter the gland because of direct communication of fistulous tract and duct (). The final diagnosis was given as persistent parotid fistula with right parotid duct secondary to chronic suppurative parotitis.\nPatient was taken under short general anesthesia; methylene blue ink was injected into the fistulous opening using a 26-gauge needle to identify the tract. An elliptical incision of 1 cm diameter was taken around the fistulous opening, which included the scar tissue. The skin island was then held with skin hooks and the subcutaneous tissue dissected until the fistulous tract containing dye was visible.\nThe fistulous tract was then traced proximally until it entered the thick parotid fascia. The fascia was incised, and the tract was traced up to the superficial lobe of the parotid. Superficial lobe of the parotid was carefully dissected, and fistulectomy was done after ligating the attachment of the fistulous tract to the Stenson’s duct (). Closure was done in two layers using vicryl 3-0 and nylon 4-0 sutures () followed by tight pressure dressing. Patient was discharged with oral amoxicillin with cloxacillin, diclofenac sodium, and cetirizine on third post-operative day. Sutures were removed on the 7th day. Regular follow-up was done for 1 year, the fistulous tract healed satisfactory () and no complain of recurrence was encountered by the patient. The salivary flow was also adequate and was periodically checked by sucking lime.
A 47-year-old female with no comorbidities presented in January 2017, with a history of bilateral pedal edema for 5 months. She had noticed a lump in her left breast in 2011, which she ignored, which was later diagnosed as carcinoma of the breast in 2015. She opted for Siddha medicine for carcinoma of the breast, which she took for 12 months starting January 2016. She developed oral ulcerations and bilateral pedal edema 9 months after initiation of the treatment. On evaluation, she was detected to have proteinuria (dipstick 3+) and microscopic hematuria (6–8 red blood cells/high power field). The laboratory results at presentation are shown in . Renal biopsy showed 20 glomeruli that were enlarged and there was mild increase in mesangial cellularity with underlying patent capillaries and uniformly thickened capillary basement membrane (BM). Eight glomeruli showed segmental endothelial proliferation with infiltration by numerous neutrophils (). Few glomeruli showed segmental sclerosis and one showed partial fibroepithelial crescent (). Immunofluorescence staining showed peripheral fine granular deposits of IgG and C3c and minimal C1q in the glomeruli. Electron microscopy (EM) study showed uniformly thickened BM and subepithelial electron dense deposits () and no electron dense deposits elsewhere, which was consistent with MN.\nBased on our previous experience with the index case, we suspected mercury poisoning causing MN, which was confirmed by the presence of very high levels of mercury in the urine (). The patient received oral prednisolone, which was discontinued after 2 weeks in view of intolerance and uncertainty about the efficacy of steroid in mercury-induced MN. She was initiated on Dimaval at a dose of 300 mg/day and the dose was increased to 400 mg/day, which was well tolerated. She received a total of 12 g of Dimaval over 6 weeks. There was a 20-fold increase in urinary mercury excretion following oral chelation therapy, confirming the efficacy of the drug. The urine mercury had reduced to an insignificant level (2.66 μg/L and 4.79 μg/day) after the completion of Dimaval therapy. There was a significant improvement in nephrotic state after chelation therapy, with improvement in serum albumin to 3.0 g/dL and proteinuria (urine PCR: 0.9 g/g of creatinine).
A 77-year-old male with a past medical history of coronary artery disease status post coronary artery bypass grafting, hypertension, chronic obstructive pulmonary disease, diabetes mellitus type 2, and cerebrovascular accident presented to a local hospital with acute abdominal pain and bloating. A computed tomography (CT) scan of the patient's abdomen and pelvis was performed and showed intraabdominal bleed and multifocal liver lesions. Initial complete blood count (CBC) revealed a hemoglobin of 7 g/dL and he was transfused one unit of packed red blood cells prior to transfer to our institution. On arrival, CT angiogram of the abdomen and pelvis showed multiple dense, heterogeneous masses throughout the liver with associated perihepatic and intraperitoneal hemorrhage and areas of tumor blush were noted but no extravasation was seen to suggest active hemorrhage. There was also multiple enlarged periportal and upper mesenteric lymph nodes, likely representing metastatic adenopathy. There were no lesions present on the pancreas. CT chest was obtained and showed no evidence of intrathoracic metastatic disease. Initial blood work revealed normal liver function tests, appropriate response in hemoglobin to transfusion and negative viral hepatitis panel. Tumor markers revealed AFP elevation to 8705 ng/mL, normal Carcinoembryonic Antigen and Cancer Antigen 19-9. Magnetic Resonance Imaging (MRI) of the abdomen and pelvis showed multiple lesions throughout the liver with targetoid appearance. There was no evidence of cirrhosis and these lesions did not have imaging characteristics of typical HCC (Figure ). It was suspected the multifocal liver lesions were HCC given the elevated AFP. However, the MRI was not consistent with HCC and a liver biopsy was obtained. Pathology results were consistent with poorly differentiated, large cell-type neuroendocrine carcinoma with metastatic disease to the liver (Figure ). The patient had an unremarkable colonoscopy and esophagogastroduodenoscopy six months prior to presentation therefore, it was suspected the primary origin of NET was in the small bowel. Regarding the intraabdominal bleed noted on initial CT scan, this remained stable on repeat scans and surgery recommended conservative management. The planned chemotherapy regimen will be Carboplatin and Etoposide.
Due to frequent headaches, epileptic seizures, speech disorders, and stiffness of the right half of the body in a 33-year-old woman, emergency imaging by a computed tomography (CT) was performed in June 2011. The scans showed a well-limited tumor mass without necrosis, located to the left in the frontal parietal-temporal region, measuring 56 × 45 × 51 mm (Fig. ). A month later, the patient underwent surgery during which a complete resection of the tumor was performed. After the inspection of a sample of tumor tissue, a diffuse astrocytoma Gr-II was diagnosed. After surgery, the patient was hospitalized in the radiology ward, where 3D conformal radiotherapy was performed using a 6 MW linear accelerator. The oncological treatment was completed by administering a therapeutic dose of 54 Gy in 27 fractions. A control scan using the nuclear magnetic resonance imaging (NMRI) technique performed in April 2012 (Fig. ) showed the presence of local recurrence, with a diameter of 5 mm, and the scan from September 2012 showed an increase of the tumor to 11 mm (Fig. ). In February 2013, due to the weakness of the right limbs and speech disturbances, an emergency NMRI was performed and found the presence of an extensive cerebral edema and recurrent tumor, 40 mm in diameter, which was spreading towards the basal ganglia, and the midsagittal plane shifted approximately 9 mm to the right (Fig. ). The patient immediately started receiving antiedema therapy and antiepileptics (dexamethasone 16 mg/day), and oncology treatment was continued with the introduction of TMZ in 6 cycles of 28 days at a dose of 200 mg/m2 of body surface area, for 5 days during each cycle. In March 2013, the patient applied for PT and began taking it along with TMZ. A combination of herbal medicines marked as phytotherapy of salvation was immediately introduced. A control NMRI performed in October 2013 showed that there had been a regression of the tumor, with the diameter being 22 mm at the time (Fig. ). The dexamethasone dose was decreased to 8 mg/day. After 6 cycles of therapy with TMZ, the patient completed oncological treatment in September 2013, and further treatment consisted solely of PT. The only pharmaceutical drugs she continued to take were antiepileptic drugs. Subsequent control scans performed in February and October 2014 showed continuous tumor regression (Fig. ). Finally, 30 months after the introduction of PT, the tumor could not be detected on the control scan from 31 August 2015, and the irregular dotted area that postcontrastly raised the signal intensity was recognized by a physician as a scar from the previous surgery from 2011 (Fig. ). This patient used herbal remedies with full capacity until there were no radiologically signs of a tumor, respectively 30 months. After this, she kept taking all of the five teas for 6 months, but every other day, which concluded the PT.
An 81-year-old Portuguese Caucasian woman, in the immediate post-operative period for incisional relapsing hernia, developed an enterocutaneous, high debit fistula requiring total parenteral nutrition.\nOur patient presented no other major medical comorbidity. Her surgical history consisted of seven abdominal operations. The first was a total hysterectomy with bilateral adnexectomy by a midline abdominal incision (1979). Secondly, two years later she developed an incisional hernia and was submitted to a herniorraphy (1981). Her third operation was a laparotomy for intestinal occlusion due to adhesions (1984). Then, three years later she developed a new incisional hernia that was corrected by hernioplasty (1991). Because of surgical site infection, the prosthesis had to be removed and replaced (1991). After a new episode of intestinal occlusion, this time with necrosis, a segmental resection of ileum (2004) was performed, after which she presented with a recurrence of the incisional hernia and was operated on again by hernioplasty (2008). There was a new recurrence of the incisional hernia one year later and an application of biological prosthesis was completed (2009).\nThe surgical team had no data about the intestinal occlusions and the following resection operations that were reported later by one of the daughters of our patient, who lived abroad. No detailed medical reports had been presented, and it seemed that some of the operations had been performed in another hospital during occasional stays of our patient at her daughter's home abroad.\nThe first approach to the central venous catheterization was made through the right subclavian vein, following the Seldinger technique. During the introduction of the wire, resistance was encountered, so the surgeon extracted the guide and reattempted introduction. During this second attempt the arterial vessel was accidentally punctured and local compression was applied to successfully stop the bleeding.\nA new approach was attempted with the same catheter on the left subclavian vein, which was successful and without complications. A control chest X-ray was ordered and showed the correct positioning of the catheter.\nThe following day, during a medical review, a hematoma on the neck of our patient located at the right supraclavicular fossa was noted, so a computed tomography (CT) contrast scan was performed. The results of the CT scan showed a small right supraclavicular fossa hematoma, with no active bleeding, and a triangular foreign body of metallic tomographic appearance, approximately 5 mm in length, in the interstitial space between right subclavian vein and artery. There was no pneumothorax or hemothorax. The left subclavian vein catheter was intact and well positioned (Figures , , ).\nWe presumed that the foreign body detected was a central venous catheter tip. This probably fragmented during the extraction of the device placed in 2004 when our patient underwent intestinal resection, and later migrated to the interstitial space between the vessels. However, we have no means to confirm our theory. The only certainty we had is that it is not a complication of our procedure since we did not change the catheter during its replacement and our patient's CT scan results showed two catheter tips.
A healthy 39-year-old woman (gravida 4, para 1) visited our hospital as an outpatient because of PPP at 33–3/7 weeks of gestation. Her previous pregnancies involved 1 CS because of intrahepatic cholestasis of pregnancy in 2005, and 2 induced abortions in 2010 and 2012, respectively. Magnetic resonance imaging showed the placenta completely covering the internal cervical orifice. Based on these findings, the patient was considered to be at high risk for PPP. She subsequently underwent antenatal monitoring at 1-week intervals until 36 weeks of gestation, and then presented to the obstetric ward.\nThe surgical bleeding volume was expected to be large and the patient had a strong desire to maintain her fertility. A planned PTUI CS was; therefore, performed under CSEA at 36–2/7 weeks of gestation. She had a prior history of difficult airway and a predicted difficult airway was suspected by the anesthesiologist, and GA was therefore excluded. On arrival in the operating room, her blood pressure was 123/72 mmHg and her heart rate was 79 beats/min. CSEA was administered with 2.2 mL heavy bupivacaine (0.5%) into the subarachnoid space. The spinal needle was removed and the epidural catheter was inserted without complications. The patient was then placed in a supine position with left uterine displacement. Spinal block reached the level of T7 at 10 minutes.\nA vertical abdominal incision was made, and the first transverse incision was then made near the uterine fundus and above the upper border of the placenta, without transecting the placenta. A healthy female infant weighing 2320 g, height 46 cm, was delivered smoothly from the first incision. The newborn's Apgar scores were 10 and 10 after 1 and 5 minutes, respectively. The placenta remained in the uterus avoiding iatrogenic partial separation, and a uterine fundal incision demonstrated minimal bleeding from the incision site, which was easily controlled by blood vessel ligation. Between delivery of the neonate and the second transverse uterine incision, 200 μg IV nitroglycerin was administered, followed by a continuous infusion of 0.3 μg/kg/min increased to 0.5 μg/kg/min until the obstetrician observed sufficient uterine relaxation. The patient's blood pressure was monitored continuously. The fundal uterine incision was sutured immediately. Shed blood from the surgical field was collected and washed using IV saline 0.9% to prepare for re-infusion. The patient's blood pressure remained at about 95–120/55–70 mm Hg throughout the 22-minutes suturing procedure, and her heart rate remained at 80 to 100 beats/min. One 6-mg bolus of ephedrine was administered for hypotension. Bilateral small openings were made in an avascular area of the ligament at the level of the cervix, and a narrow rubber tourniquet was passed through both openings and ligated tightly below the cervix to restrict uterine blood flow. The uterine body under the first fundus incision was also ligated tightly by another tourniquet to prevent bleeding from this incision. The nitroglycerin infusion was then stopped and a second transverse incision was made in the lower segment of the uterus, and the entire placenta was carefully removed under direct observation. The placenta was revealed to cover the entire anterior wall of the lower uterine segment. The placenta was completely removed and the tourniquet restricting blood flow to the uterine cervix and body was loosened, allowing confirmation of hemostasis. After confirming no obvious bleeding, the second incision in the lower segment of the uterus was closed by continuous sutures.\nThe total intraoperative blood loss was 1400 mL, and 421 mL of autologous red cells, washed with IV saline 0.9%, were re-infused back into the patient during the operation. A leukocyte filter was used to minimize the risk of amniotic fluid embolism during red cell re-infusion. The patient was transfused with crystalloid fluid (3000 mL) and hydroxyethyl starch (500 mL). Both the parturient and neonate recovered uneventfully and were discharged 4 days later.
A 66-year-old woman presented to our institute with chief complaints of difficulty in horizontal gaze and opening her mouth. She started complaining of neck and shoulder pain at the age of 58 years and had been on physiotherapy and nonsteroidal antiinflammatory drugs intake for pain control. However, at the age of 61 years, the pain worsened, and she became unable to lie down in a supine position. She had difficulty in gazing upwards at the age of 64 years. She could not keep a horizontal gaze and felt difficulty in opening her mouth because of her chin on chest position at the age of 65 years. At this point, she was diagnosed as AS at the municipal hospital and was referred to our institute for further treatment of the cervical deformity. She had no preexisting medical history, but her mother had been diagnosed as rheumatoid arthritis.\nThe patient’s neck was fixed on flexion position and was slightly rotated to the right. She could not look in front without extending her trunk. The range of mouth opening was severely restricted to approximately 2 cm because her chest was disturbing her jaw movement. She had difficulty in taking in solid food. Her upper and lower extremities had full muscle strength but had slightly restricted range of motion. She had no apparent neurologic symptoms, based on the normal deep tendon reflexes and the absence of sensory disturbances.\nA genetic test was positive for HLA-B52 and B62 but negative for HLA-B27, one of the representative genes for AS. Dual-energy x-ray absorptiometry showed osteoporosis with T score of -4.2 at L2–4 and -3.3 at the femoral neck. The Bath Ankylosing Spondylitis Disease Activity Index score was 8.4 points.\nCervical radiographs at the age of 63 years showed a C0–2 angle of 7° on flexion and 25° on extension, showing 18° of range of motion (). The middle to lower cervical spine was straight and fixed, with a C2–7 angle of -2°. Cervical radiographs at the age of 66 years, immediately before surgery, showed a C0–2 angle of 1° on flexion and 7° on extension, indicating progression of the C0–2 kyphosis for 2 years (). A radiograph of the sacroiliac joints showed ankylosis on the left (grade 3) (). Her chin-brow vertical angle was 49°. Sagittal images on preoperative computed tomography (CT) () and magnetic resonance imaging () showed basilar impression and mild compression of the medulla oblongata. However, neurological examination revealed that she was neurologically intact. Contrast CT revealed ponticulus posticus on the right ().\nAlthough 3-column osteotomy at the upper or middle cervical spine would have been effective in obtaining adequate correction, it was technically highly demanding and was expected to have high risks for perioperative neurological and vascular complications. In addition, C0–1 seemed to be mobile and could expect a slight correction, which might slightly improve the chin on chest deformity. Considering the safety, we planned fixation surgery at C0–5 posteriorly to prevent kyphosis progression, with slight correction of kyphosis at C0–2.\nFor general anesthesia, fiberoptic visualization was used to facilitate nasotracheal tube insertion to secure airway access. The patient was placed in a prone position with 2 kg of horizontal traction to stabilize the head position. The occipital plate was placed, followed by placement of the pedicle screw only on the right side of C2. A lamina screw was placed on the right side of C2, because of the high riding vertebral artery on the left side. Lateral mass screws were placed on both sides of C3, C4, and C5. Thereafter, over lordotically contoured titanium rods with a diameter of 3.5 mm were fixed on both sides of the occipital plate, and the correction was performed by gradually pushing down the bilateral rods onto the C3–5 screws using the cantilever technique. The rods were contoured to create lordosis along C0–2. We did not perform any osteotomies to the posterior elements for the kyphosis correction. Although there was resistance from the rod rigidity, we felt neither palpable clacking nor any apparent motions at the facet joints at C3/4.\nAn intraoperative radiograph after fixing the rods on the C3–5 screws revealed that the C2–7 angle was corrected to 28°. However, there was apparent anterior opening of the vertebral bodies between C3 and C4 (). There were no changes in the vital signs, including blood pressure and pulse rate, and no abnormality in the motor evoked potential responses. Eventually, the surgery was finished after local bone grafting.\nAfter surgery, the patient’s consciousness and limb movements appeared to be normal, even while intubated. Notably, the postoperative CT images showed massive swelling anterior to the C3/4 level; this suggested hemorrhage from the fracture site (). Massive swelling around the pharynx made postoperative extubation difficult; therefore, tracheotomy was performed.\nA solid bone fusion between the C3 and C4 was expected to take a long time, and the stability of the lateral mass screws on C4 and C5 with lacking anterior column support at C3/4 was considered unreliable to maintain the correction. External fixation by bracing would be another option. However, C3/4 had no anterior support, and we could not be sure that the brace would be enough to support the cervical spine until obtaining solid bone fusion, since the stability of C4 and C5 lateral mass screws were unreliable. Thus, 7 days after the first surgery, a staged surgery was performed to stabilize the C3/4 by extending the fusion to T3 to prevent postoperative complication at cervicothoracic junction, and to obtain bone fusion by grafting an additional iliac bone. One month after surgery, the swelling at retropharyngeal space was recovered (), and the tracheostomy cannula was removed. At 1.5 months after surgery, videofluoroscopy revealed normal swallowing, and she was able to start food intake.\nAfter the surgery, she was able to look in front, breathe easily, and eat solid food without difficulty. Two years after surgery, the radiograph showed C0–2 angle of 10° and C2–7 angle of 40° without any correction loss (). Her chin-brow vertical angle was corrected to 17°.

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