Uncommon Course Research Articles

Common Illness with an Uncommon Course- In Adolescents use HEADSS

Adolescence is a period of emotional instability, during which high risk behavior including experimentation with drugs is common. Clinical manifestations of substance abuse and its withdrawal may mimic common illnesses. We report an adolescent who presented with acute dysentery with unusual course. HEADSS screening revealed psychosocial stressors leading to cannabis addiction. The child was diagnosed to have cannabis withdrawal. He was referred to the de-addiction clinic. HEADSS is a useful tool to identify risk factors and protective factors in adolescents. HEADSS screening during each interaction and early intervention when needed must be an essential component of adolescent health care.

Uncommon course of a common disease: An interesting case of guillain–Barre syndrome

Uncommon course of a common disease: An interesting case of guillain–Barre syndrome

A simple appendicitis? An anatomical pitfall

Rationale: We present a case of appendicitis with an uncommon course due to rare anatomical location of the appendix in the right retroperitoneal space below the diaphragm and above the liver. Patient's concern: A 32-year-old, previously healthy male with a history of congenital diaphragmatic hernia repair in childhood, presented with 3 days of mild, colicky, central abdominal pain associated with fever, nausea and vomiting. At presentation, pain was localized to the right lower quadrant. Diagnosis: Even though diagnosis of appendicitis was clear, we decided to confirm it with computer tomography (CT). CT revealed elevation of the right dome of the diaphragm and perforated appendix located above the liver. Intervention: Appendectomy was performed via right subcostal approach instead of usual incision in the right lower quadrant. Outcome: Patient recovered well and was discharged on the 5th day after operation. Lessons: Previous congenital diaphragmatic hermia repair may change the location of the appendix. The appendix at rare locations could lead to an uncommon course of appendicitis. On this very note, surgeons should have a high index of suspicion, and CT may help avoid inadvertent complications.

Pituitary Macroadenoma Masked by Iatrogenic Adrenal Insufficiency: A Diagnostic Blind Spot

Background: Exogenous steroid use is the most common cause of central adrenal insufficiency. Depending on the duration and strength used, it may take months to years for the hypothalamic-pituitary-adrenal (HPA) axis to recover after the steroid is stopped. We report a case of iatrogenic hypoadrenalism with persistent suppression of the HPA axis for 13 years, discovered later to be due to a second pathology. Case: A 48 year old lady presented in 2005 with weight gain of 20 kg over 1 year and florid Cushingoid features. 9AM cortisol was undetectable (<12 nmol/L). History taking revealed use of oral dexamethasone at various dosages over the past 9 years for her knee pains. A diagnosis of iatrogenic adrenal insufficiency was made. She was started on hydrocortisone replacement, and was advised to stop the over-the-counter steroids. By 2011 her short Synacthen test (SST) showed much improved functioning of the HPA axis (cortisol 182 (0 min) -> 329 (30 min) -> 408 nmol/L (60 min) [N peak>500 nmol/L]), and she was back to her usual body weight. However, subsequent monitoring revealed declining trend of 9AM cortisol from 135 nmol/L (2013) -> 99 nmol/L (2014) -> 57 nmol/L (2015) -> 64 nmol/L (2016) -> 18 nmol/L (2017) [N 166–507 nmol/L]. Hydrocortisone compliance and abstinence from exogenous steroids was confirmed with the patient. The ongoing hypofunction of the HPA axis was continually attributed by multiple physicians to her history of prolonged use of dexamethasone. In 2018, at the age of 60, the lady presented with new onset headaches, blurred vision, and bitemporal hemianopia for 3 months. MRI showed a 1.8x1.8x3.5 cm (WxAPxH) pituitary mass with suprasellar extension compressing the optic chiasm. Blood tests revealed panhypopituitarism: SST cortisol 17 -> 59 -> 49 nmol/L, ACTH 2.7 pmol/L [N <10.1 pmol/L]; fT4 9.5 pmol/L [N 12–22 pmol/L], TSH 0.97 mIU/L [N 0.27–4.2 mIU/L]; LH <0.1 IU/L, FSH 0.52 IU/L (menopause at age of 48); IGF-1 27 µg/L [N 41–279 µg/L]; prolactin 17 mIU/L [N 102–496 mIU/L]. After partial excision of the mass her vision improved, but remained dependent on hydrocortisone and thyroxine supplements. The lesion was pathologically proven to be a pituitary macroadenoma. Discussion: This case presents the uncommon course of a patient who had almost recovered from iatrogenic hypoadrenalism, only to lapse back into worsened central adrenal insufficiency, as part of panhypopituitarism related to an undiagnosed pituitary mass. In retrospect, the unusually protracted state of hypocortisolemia and the atypical waxing and waning HPA axis should have alerted one to consider alternative etiologies at work. As LH-FSH and GH deficiencies commonly develop before ACTH and TSH deficiencies in most pituitary macroadenomas, a lower threshold for testing the other anterior pituitary hormones followed by imaging of the pituitary could have picked up the tumor earlier in this patient.

Impact of Osteophytectomy on Swallowing Function in a Patient with Chronic Dysphagia

Anterior cervical osteophytes are common in the geriatric population. Dysphagia can occur in individuals suffering from these spinal abnormalities. Surgical intervention is an uncommon course of treatment for these patients, but is often utilized as a last resort with the hope of swallow recovery. The purpose of this article is to highlight a unique case study documenting the required treatment course for dysphagia associated with osteophytes and subsequent osteophytectomy. We review current literature of both conservative and surgical interventions, as well as discuss the rehabilitation course for a complex patient with persistent dysphagia. Various outcome measures were utilized during the patient’s inpatient stay to track progress including the Functional Oral Intake Scale (FOIS), the Bolus Residue Scale (BRS), Penetration Aspiration Scale (PenAsp), Dysphagia Outcome Severity Scale (DOSS), and a Modified Barium Swallow Study (MBSS). The patient received rehabilitative training including oral motor and pharyngeal strengthening exercises, respiratory strengthening, speech instruction, cognitive retraining, and instrumental assessment. Following osteophytectomy and dysphagia rehabilitation the patient did not show any change based on a repeat MBSS which revealed the necessity for the patient to remain nothing per oral (NPO). The patient demonstrated an inability to manage his secretions, requiring continual suctioning. Upon discharge the patient remained NPO with the exception of ice chips, utilized a PEG for nutrition, and had a red capped tracheostomy. He was on room air and independently utilized oral suction as needed for secretion management. Our patient’s clinical course was not aligned with typical osteophytectomy recovery as progress after his 25-day inpatient stay was limited. The goal of this case study is to contribute information to the limited and variable data available regarding treatment options, outcome measures and timelines for recovery as it pertains to patients who undergo an osteophytectomy.

Neutrophilic Dermatosis in Pregnancy: An Uncommon Course.

Pyoderma gangrenosum (PG) is a neutrophilic-mediated inflammatory skin disease characterised by the rapid onset of painful, hemorrhagic pustules developing into necrotic ulcers occurring predominantly in women aged 20-50 years. According to the literature, all patients reported no change or worsening of the disease during pregnancy. We herein present the case of a 34-year-old woman that developed a neutrophilic dermatosis of the hand reporting complete resolution of the skin disease during pregnancy.

(71) An uncommon course of fibrocartilaginous embolism causing acute spinal cord infarction following trampoline-related injury.

Introduction: Non-traumatic acute onset paralysis, particularly due to a fibrocartilaginous embolism (FCE), is exceedingly rare in the pediatric population. FCE has become more popular in the literature and until recently, most of the cases have been diagnosed post-mortem and survivors have generally had poor outcomes. This is a case of a child presenting with acute ischemic spinal cord infarction with presumed FCE following trampoline jumping who had a remarkable recovery. Case Description: A 10-year-old previously healthy male presented with acute onset quadriplegia, apnea and altered mental status. Approximately 5 hours …

(71) An uncommon course of fibrocartilaginous embolism causing acute spinal cord infarction following trampoline-related injury.

(71) An uncommon course of fibrocartilaginous embolism causing acute spinal cord infarction following trampoline-related injury.

Perforation of the gallbladder into the abdominal wall: A rare manifestation of biliary disease

A 54-year-old man was admitted to our surgery department complaining of right upper quadrant abdominal pain. The working diagnosis of acute cholecystitis was based on the clinical picture, laboratory tests, and ultrasound findings. Because of the uncommon course of the disease, computed abdominal tomography was performed, showing multiple stones in the anterior abdominal wall. On surgery, the fistula, containing stones and pus connecting the gallbladder and the abdominal wall muscles, was found.

Uncommon course of obturator nerve through an osseus tunnel: Clinical relevance

A unilateral variation in the course of the left obturator nerve was observed during cadaveric dissection of the pelvis and lumbar plexus. The donor was a 94‐year‐old Caucasian male died of renal and respiratory failure. Typically, obturator nerve arose from the ventral rami of lumbar spinal segments 2–4 and traveled forwards along the lesser pelvic lateral wall on the obturator internus to the obturator foramen. The variant obturator nerve descended normally anterior to sacroiliac junction continued the lateral pelvic course and entered a 6 cm long osseus tunnel in the superior rami beginning at the iliopectineal junction (close to ischial spine) and emerged to continue a normal course joined by the obturator vessels. The right side exhibited normal course. Although the course of the obturator nerve is often consistent, variations in pelvic vascular and nervous structures do exist, and clinicians should be aware of the possibility of encountering less common presentations of the obturator nerve. Knowledge of alternative course of obturator nerve has a significant impact during pelvic surgery, addressing pelvic fractures resulting from trauma or osteoporosis and approaches to regional anesthesia. This is the first report on the uncommon course of obturator nerve through an independent osseus tunnel in the pelvic bone. Its clinical relevance in surgery or trauma related to pelvic bone will be discussed.

“Unsteady Gait”: An Uncommon Presentation and Course of Malignant Melanoma in Terminal Ileum—A Case Report and Review of Literature

Malignant melanoma within the gastrointestinal tract is an uncommon neoplasm that is usually metastatic in origin, with primary melanomas being relatively uncommon. Embryologically melanocytes normally exist in the esophagus, stomach, small bowel, and anorectum and this theory supports the primary melanoma of the gastrointestinal tract that has been confirmed for lesions occurring through several published reports. However, most patients with brain metastases from malignant melanoma are diagnosed after treatment for known extracranial metastases and have poor outcomes. Our case is unique in that we discuss an unusual case of 69-year-old female patient presented with unsteady gait as the first symptom of disease and where the presumed primary lesion later was found in the terminal ileum on colonoscopy. Treatment consisted of surgical removal of the terminal ileal lesion with chemotherapy, whole-brain radiotherapy, and cyberknife radiosurgical procedure. Patient was in remission for more than 14 months and later succumbed to disease. Despite the advances in therapeutic options, prognosis for patients with melanoma brain metastases remains poor with a median survival time of six months after diagnosis.

Invited Commentary: Left Isomerism or “Isolated Atrial Inversion”?

In the accompanying article, Chenu and colleagues describe "an uncommon course of the right superior vena cava in a patient with heterotaxy syndrome." In their discussion, they pose the question, "Is this an isolated atrial inversion, or a case of left isomerism?" They proceed to comment "this remains a debate among cardiac morphologists." I can agree with them on their first description, since although I have previously examined many hearts from patients with isomerism of the left atrial appendages, or "polysplenia syndrome," I had not noticed the unusual feature they have emphasized in their case report; although, as I will show, the evidence was there for me to observe their feature of emphasis. They deserve great credit, therefore, for bringing this feature to our attention. I also agree with their first statement, namely that their patient has the left isomeric variant of the so-called visceral heterotaxy, but I must question their suggestion that the patient may exhibit mirror-imaged arrangement of the atrial chambers or "atrial situs inversus." I would question even more strongly their comment that the distinction remains "a debate among cardiac morphologists." As I will further show, the distinction has major clinical implications, not only for pediatric cardiac surgeons but also for those involved with genetic counseling.

An Uncommon Course of the Right Superior Vena Cava in a Patient With Heterotaxy Syndrome

We present the case of an infant with congenital heart disease which includes a partial atrioventricular canal defect with the absence of the atrial septum (common atrium) and an extremely uncommon course of the right-sided superior vena cava (SVC) including an intra-atrial segment coursing intramurally along the right posterolateral atrial wall, with an intracardiac orifice situated low within the right side of the atrium, close to the atrial orifice of the right hepatic veins. This feature of the anatomy was discovered intraoperatively at the time of surgical repair. Systemic venous anatomy also included interrupted inferior vena cava (IVC) with azygos continuation to a left-sided SVC draining directly into the left side of the atrium. The successful surgical procedure included tunneling of the left-sided SVC to the right side of the common atrium and atrial septation with a patch.

Uncommon Course of the Ulnar Artery

Uncommon Course of the Ulnar Artery

CD46-associated atypical hemolytic uremic syndrome with uncommon course caused by cblC deficiency

CD46-associated atypical hemolytic uremic syndrome with uncommon course caused by cblC deficiency

Successful Transseptal Puncture and Cryoballoon Pulmonary Vein Isolation in Adverse Anatomy

A 56-year-old male patient was admitted for catheter ablation of highly symptomatic paroxysmal atrial fibrillation. During the procedure, a rare anatomic variation with an interrupted inferior caval vein (ICV) branching suprarenal into an azygos venous continuation draining into the superior caval vein was detected. Using angiography, a continuation of the ICV leading into the liver, subdividing into 5 smaller branches draining into the right atrium (RA), was displayed. Despite this challenging anatomic variation, successful pulmonary vein isolation (PVI) was performed. A 56-year-old male patient was admitted for catheter ablation of highly symptomatic drug-refractory paroxysmal atrial fibrillation (2 to 3 episodes per week), first diagnosed 48 months before the procedure. Physical examination revealed no anomalous findings, and medical history was unremarkable except for a known 1-vessel coronary artery disease with status post bare metal stent implantation in the left anterior descending artery, normal left ventricular function, and left atrial (LA) diameter of 51 mm. No LA thrombus was detected before the procedure by transesophageal echocardiography. No additional preinterventional imaging was performed. ### Ablation Procedure After written informed consent was obtained, the procedure was performed under conscious sedation using boli of midazolam, fentanyl, and a continuous infusion of propofol (1%). Venous accesses (8F) were achieved via the right femoral vein, the left femoral vein, and the left subclavian vein. Positioning of both deflectable diagnostic catheters (7F, Webster, Biosense Webster, Diamond Bar, Calif) in the coronary sinus from the left subclavian vein and at the His-bundle region via the left femoral vein was unremarkable. ### Transseptal Puncture The guide wire for the first transseptal sheath was introduced via the right femoral vein. The guide wire exhibited an uncommon course and did not enter the RA from inferior; angiography revealed a persistent vena azygos draining into the superior caval vein (Figure 1). An additional angiography from the ICV demonstrated suprarenal …

Sudden ST elevation with angina-like pain in myocarditis. An uncommon course of a common disease: strategic role of cardiac magnetic resonance

A case of a young man with myocarditis simulating acute coronary syndrome is reported. The possibility of vasospasm is discussed. The use of cardiac magnetic resonance imaging (MRI) is highlighted.

Uncommon course for a vertebral artery dissection: Rupture, occlusion and recanalization

Intracranial arterial dissections of the vertebrobasilar system are recognized as a cause of stroke. Although the pathogenic mechanism underlying this phenomenon is unknown, in some cases the stroke originates from subarachnoid hemorrhage, while in others ischemia is the cause. In cases where hemorrhage occurs, occlusion of the lesion is effective in reducing the risk of re-bleeding. However, deciding on treatment is difficult in uncommon cases in which occlusion occurs immediately after hemorrhage. Intracranial arterial dissections of the vertebrobasilar system often present as subarachnoid hemorrhage, and the most appropriate surgical treatment remains controversial. In this report, we describe a rare case of vertebral artery dissection in which occlusion occurred immediately after subarachnoid hemorrhage. Serial angiography revealed spontaneous recanalization of the right vertebral artery dissection, as well as enlargement. The progressive angiographic changes of the ruptured vertebral artery dissection and the endovascular treatment of such arterial dissections are discussed.

An uncommon presentation and course of metastatic malignant melanoma: a case report

Most patients with brain metastases from malignant melanoma are diagnosed after treatment for known extracranial metastases and have a poor outcome despite various local and systemic therapeutic approaches. Here we discuss an unusual case where a 45-year old patient presented with a brain metastasis as the first symptom of disease and where the presumed primary lesion later was found in the gastro-intestinal tract. Treatment consisted of sequential surgical removal of a total of 4 tumor sites (2 extracranially), whole-brain radiotherapy and two radiosurgery procedures within 13 months. Following her last treatment, the patient has now been in remission for 20 months. This case illustrates that some patients with multi-organ melanoma manifestations may benefit from the repeated use of effective local therapeutic approaches and may experience a quite favourable prognosis.

Reduced TH-1 Cytokine Release in an Adult Patient with Chronic Relapsing Mycobacterium malmoense Infection

An unusual course of infection with Mycobacterium malmoense is described in a patient receiving chronic but mild immunosuppressive therapy for rheumatoid arthritis. Symptoms mimicking Crohn's disease deteriorated under intensified immunosuppression and surgery. Judging from the patient's course under treatment specific for M. malmoense, the gastrointestinal symptoms were rather manifestations of a chronic relapsing mycobacterial infection. Detailed immunological investigation of the patient revealed a severely impaired TH-1 cytokine response as the immunological background for this uncommon course.